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American Journal of Clinical Medicine® Owned and Published by the American Association of Physician Specialists, Inc. Peer Reviewed. Listed in Google Scholar

Summer 2014 • Volume Ten, Number Two

FEATURED IN THIS ISSUE 56

The Utility of Anti-tissue Transglutaminase Antibody-IgA (tTG-IgA) Testing for Celiac Disease in Adults with Down Syndrome

Comparison of Actual and Predicted Emergency Department Wait Times

Factors Affecting Rural Patients’ Compliance with Screening Colonoscopy: Importance of Distance and Physician

A Case Report on the Recurrence of Lemierre’s Syndrome: Awareness to Diagnosis and Treatment

Extraosseous Ewing Sarcoma of the Larynx: A Report of a Rare Case

AJCM

HEALTH CARE POLICY REVIEW

The Declining Number of Family Physicians Practicing Obstetrics: Rural Impact, Reasons, Recommendations and Considerations Daniel M. Avery, Jr, MD John T. McDonald, Jr, MD, CPA

MEDICAL ETHICS

Medical Ethics Without the Rhetoric Mark Pastin, PhD

CASE REPORTS

In This Issue

ORIGINAL RESEARCH The Utility of Anti-tissue Transglutaminase Antibody-IgA (tTG-IgA) Testing for Celiac Disease in Adults with Down Syndrome

Caleb A. Awoniyi, MD, PhD Douglas Hanson, MD

56 87

LouAnn H. Woodward, MD Jason Zimmerman, BSN Kristen C. Isom, RN Richard L. Summers, MD

Factors Affecting Rural Patients’ Compliance with Screening Colonoscopy: Importance of Distance and Physician Arsheeya Mashaw, MD Justin T. Arcury, MS

Extraosseous Ewing Sarcoma of the Larynx: A Report of a Rare Case Nicole F. De Simone, DO, MPH, Capt, USAF Anuj Parikh, DO Kurt Graupensperger, DO James P. Malone, MD

Brian Chicoine, MD Shalin Parikh, MD Suela Sulo, MSc Laura Chicoine

Comparison of Actual and Predicted Emergency Department Wait Times

A Case Report on the Recurrence of Lemierre’s Syndrome: Awareness to Diagnosis and Treatment

Undulant Fever in ED: A Case Report Andrzej Skarpetowski, MD

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

The Utility of Anti-tissue Transglutaminase Antibody-IgA (tTG-IgA) Testing for Celiac Disease in Adults with Down Syndrome Brian Chicoine, MD Shalin Parikh, MD Suela Sulo, MSc Laura Chicoine

Abstract Objectives: We assessed the diagnostic accuracy of the antitissue transglutaminase IgA antibody (tTG-IgA) test compared to esophagogastroduodenoscopy (EGD) in adults with Down syndrome (DS). Methods: Retrospective chart review of 152 adult patients with DS who were diagnosed with celiac disease (CD). Results: Of these patients, 141 (92.8%) had a positive tTG-IgA and 11 (7.2%) had a negative result. Eighty-one (53.3%) did not have an EGD performed due to patient or legal representatives declining. Of the 71 who had a biopsy, the result of the tTG-IgA in 29 (40.8%) was a true positive, 35 (49.3%) a false positive, 2 (2.8%) a true negative, and 5 (7.0%) a false negative. The sensitivity was 85%, specificity 5%, positive predictive value 45%, and accuracy 43.7%. Conclusions: The low specificity, accuracy, and positive predictive values of tTG-IgA in people with DS have significant clinical and financial implications in diagnosing, treating, and screening for CD in people with DS.

Introduction Celiac disease (CD) is an autoimmune reaction to the protein gluten that can damage the villi of the small intestine and interfere with the absorption of nutrients. When left untreated, CD

can lead to malnutrition, anemia, osteoporosis, liver diseases, and cancer (particularly non-Hodgkin’s lymphoma).1,2 Symptoms include abdominal bloating and pain, chronic diarrhea, vomiting, constipation, weight loss, anemia, fatigue, bone or joint pain, arthritis, bone loss or osteoporosis, seizures, depression, anxiety, canker sores in the mouth, and dermatitis herpetiformis.1 During the typical diagnostic process of CD, an antibody blood test, such as the anti-tissue transglutaminase IgA antibody (tTG-IgA) test, is performed. Positive antibody blood tests are confirmed through the “gold standard,” duodenal biopsy, performed during an esophagogastroduodenoscopy (EGD).3 TTGIgA testing has been studied extensively and the sensitivity and specificity for untreated CD has been reported to be 95%.4,5 Upon making the diagnosis, treatment consists of eating a diet free of gluten. Apart from requiring significant patient education, motivation, and follow-up, following a gluten-free diet for a lifetime can be challenging due to the high prevalence of gluten-containing foods.6 Furthermore, a gluten-free diet costs more than an average gluten-containing diet.7 Therefore, an inaccurate diagnosis can cause unnecessary challenges in both dietary compliance and financial cost. The need for an accurate diagnosis is particularly important in people with Down syndrome (DS) as a subpopulation with a higher incidence of CD.8-11 DS is the most common known chromosomal cause of an intellectual disability. It is associ-

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

ated with a higher incidence of some health problems such as hypothyroidism, type 1 diabetes mellitus, and congenital heart disease and a lower incidence of others such as atherosclerotic disease, hypertension, and solid tumors.12,13 Despite the higher incidence of CD in people with DS, studies of the diagnostic accuracy of antibody tests have primarily emphasized individuals in the population without DS6, and less is known about their accuracy in individuals with DS. Although there is lack of information regarding the accuracy of tTG-IgA in people with DS, some patients with DS and their families may elect to proceed to a gluten-free diet based on the blood test results and avoid undergoing an EGD with biopsy. Because anesthesia complications are more common in people with DS14, patients with DS or their families may elect to avoid the biopsy because it requires anesthesia; thus relying on an unsubstantiated and potentially inaccurate diagnosis of CD. An improved understanding of the accuracy of tTG-IgA is needed to assist people with DS and their families to make the decision regarding proceeding with or foregoing the EGD with biopsy and proceeding with the diet. This study examined the diagnostic accuracy of the tTG-IgA test compared to EGD with biopsy in adults with DS. The social effects and financial costs of adopting a gluten-free diet based on the tTG-IgA blood test alone are also discussed.

Methods Study Design: This is a retrospective chart review study of 152 adult patients with DS who received care for CD at the Advocate Medical Group Adult Down Syndrome Center located in Park Ridge, Illinois, from January 1, 2000, to October 31, 2012. This study was approved as expedited by the Advocate Health Care’s Institutional Review Board on December 10, 2012. A waiver of consent was granted for accessing patients’ records to collect information about their age, gender, tTG-IgA, and EGD with duodenal biopsy results. Analysis: Due to lack of evidence about the diagnostic characteristics of the tTG-IgA as compared to the EGD with duodenal biopsy in the population with DS and the exploratory purpose of the study, we relied on a convenience sample of all patients with DS who were diagnosed with CD at our institution during a 12-year timeframe. Descriptive statistics (means, standard deviations, range) are reported for age and frequencies (%) for gender and test results. Sensitivity, specificity, positive and negative predictive value, prevalence, and accuracy of the tTG-IgA test were calculated. The data for this study were analyzed using SPSS for Windows, version 20.0 (SPSS Inc., Chicago, IL).

Results There were 152 patients who met the study’s inclusion criteria. The patients were adults with a mean age of 42.3 (± 12.1, range 20-64); 77 (50.7%) were female and 75 (49.3%) were male. Of these patients, 141 (92.8%) had a positive tTG-IgA result, while 11 (7.2%) had a negative result. However, only 71 (46.7%) pa-

tients had an EGD performed. The EGD results were positive for 34 (22.4%) and negative for 37 (24.3%), while 81 (53.3%) did not have an EGD performed due to patient or legal representatives declining (Table 1). Table 1: Patients characteristics and test results (n = 152)

Variable Age, mean (SD), range

Value 42.3 (12.1) [20-64]

Gender, n (%) Male

75 (49.3)

Female

77 (50.7)

tTG-IgA Result, n (%) Positive

141 (92.8)

Negative

11 (7.2)

EGD Result, n (%) Positive

34 (22.4)

Negative

37 (24.3)

Declined

81 (53.3)

tTG-IgA = anti-tissue transglutaminase immunoglobulin A; EGD = esophagogastroduodenoscopy

Of the patients who underwent an EGD with biopsy, 29 (40.8%) had a true positive result, 35 (49.3%) had a false positive result, 2 (2.8%) had a true negative result, and 5 (7.0%) had a false negative result. The sensitivity of the tTG-IgA was 85%, specificity was 5%, positive predictive value was 45%, negative predictive value was 29%, accuracy was 43.7%, and prevalence was 47.9% (Table 2). Table 2: Diagnostic values of the tTG-IgA (n = 71)

Variable

Value

True Positive, n (%)

29 (40.8)

False Positive, n (%)

35 (49.3)

True Negative, n (%)

2 (2.8)

False Negative, n (%)

5 (7.0)

Sensitivity, %

Specificity, %

Positive Predictive Value, %

Negative Predictive Value, %

Accuracy, %

43.7

Prevalence, %

47.9

tTG-IgA = anti-tissue transglutaminase immunoglobulin A

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

Discussion The assessment of CD includes evaluating symptoms, physical exam, blood testing, and EGD with biopsy. During the EGD, at least 2-3 biopsies of the duodenum are taken as the standard diagnostic approach used at our institution for the diagnosis of CD in adults with and without DS. The most common histological findings expected include villous atrophy, crypt hyperplasia, thickening of the basement membrane under the surface epithelium, increased intraepithelial lymphocytes, and influx of immune cells in the lamina propria with enterocyte changes.15 Although the incidence of CD is higher in people with DS, the presentation of symptoms of CD is similar for people with and without DS.16 One exceptional note is the higher likelihood of adults with intellectual disabilities, including DS, of presenting with behavioral changes in response to physical health conditions.17 Therefore, in the diagnostic assessment for CD in adults with DS, both their psychological and physical symptoms should be carefully considered by both primary care and specialty physicians so that health problems like celiac disease are not missed and/or under-assessed. The next step in the diagnostic approach is blood testing, and tTG-IgA is the preferred test.6 The recommendation for tTGIgA as the preferred test is partly based on the reported high sensitivity and specificity in the general population.4,5 However, the present study found much lower values for these statistical measures of tTG-IgA in people with DS, thus making the test less accurate in the diagnostic process. However, without the findings of the present study of a low specificity and thus, an assumption of the same high specificity noted for those without DS, many patients with DS and their families opted to proceed with the gluten-free diet without a biopsy. With a positive predictive value of only 45%, more than 50% of the individuals who elected to proceed with the diet without biopsy may be on the diet despite not having CD. To proceed with the diet without a diagnosis of CD has the potential to cause unnecessary cost. The cost of gluten-free food is $1.10 more per 100 grams.7 Since an average individual eats 420 grams per day18, the increased cost is $4.62 daily and $1,686.30 per year. The cost may be further increased because in order for a person with DS to follow the diet, our clinical experience has found that the whole household may need to follow a gluten-free diet. In a family of three (including the person with DS), the potential annual cost increase may thus be over $5,000.00. If the diet is followed for 30 years by this family, the potential cost increase of the food alone is over $150,000.00. While the cost difference of gluten-free food may decline over time, presently, the cost differential can be substantial over a lifetime. In addition to the financial cost, there is potential social cost. Increased independence is a social goal for people with DS19 and this may be hampered by the inability of some individuals with DS to manage a gluten-free diet independently. As a result, inaccurate diagnosis may increase the number of individuals whose efforts at independence are unnecessarily imposed.

The issue of screening also arises. At present, there is not a recommendation to screen all people with DS with blood testing. However, while the American Academy of Pediatrics health guidelines for children with DS recommends assessing for symptoms at annual visits in early childhood20, others have recommended universal serology screening.16 One argument for screening is to prevent lymphoma of the gastrointestinal tract. Swigonski et al21 found that the cost of universal serology screening of children with DS was more than $500,000.00 per life-year gained from lymphoma and almost $5 million to prevent a single case of lymphoma. This cost was based on their use of a positive predictive value of 93%. However, with our finding of a positive predictive value of less than half of that, the potential cost of prevention of lymphoma in screening could double. The overall cost of annual universal screening for people with DS would also be substantial. There are an estimated 250,000 people with DS in the United States.22 At our hospital, the cost of the tTG-IgA blood test is $141.00. If all 250,000 were screened in a year, the cost of the blood test alone would be $35,250,000.00. However, this cost could decrease as some individuals who were diagnosed with CD would no longer need annual screening. Another substantial excess cost of using a test with a low sensitivity would be in the follow-up EGDs. Using the incidence of CD in DS of 15% and the number of people with DS as 250,000, theoretically, 37,500 people with DS will be diagnosed with CD. However, even if we assume those with a false positive tTG-IgA underwent only one biopsy in a lifetime, with a positive predictive value of 45%, an additional 45,833 individuals with a false positive tTG-IgA would require a biopsy that would ultimately be negative. With a national average cost of an EGD estimated to be $2,700.0023, the total cost of 83,333 EGDs is estimated to be $224,999,100.00. The cost of the additional 45,833 EGDs for those with a false positive tTG-IgA would be $123,749,100.00. It is important to note that the actual cost for people with DS is likely to be higher due to the frequent need for greater anesthesia, difficulty complying with testing, the potential for increased anesthesia complications, and the potential cost of treating those complications.14 The American College of Gastroenterology (ACG) guidelines for evaluating and treating CD include tTG-IgA as the recommended serology test for CD.6 High sensitivity and specificity of tTG-IgA in people without DS is important in recommending this test. However, even with a highly accurate tTG-IgA, the recommendation for the assessment of CD still includes evaluation of symptoms, serology testing, and EGD with biopsy for diagnosing CD. The need for assessing all three components of the evaluation (symptoms, tTG-IgA, and EGD with biopsy) is increased when one component is less accurate such as tTGIgA in people with DS. Another reason to assess all three components is the complicating issue of gluten sensitivity. There is a group of individuals whose tTG-IgA and biopsy are not consistent with CD but have gluten sensitivity, and who may benefit from a gluten-free diet.6 When assessing symptoms in these individuals, the ACG recommends a full evaluation in-

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

cluding serology evaluation and EGD with biopsy to rule out CD before proceeding with a gluten-free diet. Less is known about gluten sensitivity in people with DS. While celiac disease has been strongly linked to human leukocyte antigens (HLA) located on chromosome 6, with 85% to 100% of people with CD carrying either a HLA-DQ2 or HLA-DQ8 heterodimer, these tests are not part of the standard diagnostic approach to CD.24,25 Although HLA-DQ2 or HLADQ8 have been found to have better specificity than tTG-IgA, to date, no studies have assessed their diagnostic characteristics in people with DS. Also, genetic tests like HLA-DQ2 or HLA-DQ8 confirm the genetic predisposition rather than the presence of the celiac disease, and they are usually expensive and not covered by insurance, thus incurring additional financial costs. Until the diagnostic characteristics of HLA-DQ2 or HLA-DQ8 in people with DS are confirmed, EGD with biopsy remains the gold standard for making an accurate diagnosis. This study found a lower sensitivity, specificity, and positive predictive value for tTG-IgA in patients with DS when compared to those without DS.5 The importance of assessing symptoms and performing an EGD with biopsy to make an accurate diagnosis is heightened when the tTG-Ig is less accurate. Furthermore, the low values for these measures of test accuracy will limit tTG-IgA as a potential screening tool for asymptomatic people with DS. The overall financial, social, and medical costs are substantial and need to be considered when screening for asymptomatic CD in people with DS. This study has limitations that should be carefully considered when interpreting the study results. First, we relied on a convenience sample of patients with DS who were diagnosed with CD at our institution during a given timeframe, thus limiting the generalizability of the study findings to other patients with DS. Second, we relied on retrospective data that were collected through patients’ charts, thus limiting the amount of information collected for individual patients. Taking into account these two important limitations, we recommend that future studies employ prospective study designs with larger sample sizes to confirm the diagnostic characteristics of the tTG-IgA in adult patients with DS. Further research should also include evaluation of a protocol that carefully assesses pre-treatment symptoms, tTG-IgA or other serology, EGD with biopsy, and post-treatment symptoms in people with DS. It is important, however, to carefully take into consideration the feasibility, practicality, and ethics of the prospective study designs when conducting research with adults with DS.26 Finally, additional studies should include assessment of the potential need and benefit of universal screening as well as cost issues, and possibly, alternative serologic tools needed in assessing CD in adults with DS.

Acknowledgement We would like to thank Inna Trey, MD, for her help with data collection. Previous Presentations: Portions of this project have been presented as a poster at the American Academy of Family Physi-

cians Scientific Assembly in San Diego, CA, in September 2013; at the Family Medicine Midwest - Illinois Association of Family Physicians in Milwaukee, WI, in October 2013, and at the 2013 Advocate Research & Innovation Forum at Advocate Lutheran General Hospital in Park Ridge, IL. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Brian Chicoine, MD, is the medical director of Advocate Medical Group Adult Down Syndrome Center and a faculty member of Advocate Lutheran General Hospital Family Medicine Residency Program in Park Ridge, IL. Shalin Parikh, MD, is a resident physician at the Advocate Lutheran General Hospital Family Medicine Residency in Park Ridge, IL. Suela Sulo, MSc, is the patient centered outcomes research manager at James R. & Helen D. Russell Institute for Research & Innovation, Advocate Lutheran General Hospital in Park Ridge, IL. Laura Chicoine is a research assistant at Advocate Medical Group Adult Down Syndrome Center in Park Ridge, IL.

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21. Swigonski NL, Kuhlenschmidt HL, Bull MJ, Corkins MR, Downs SM. Screening for celiac disease in asymptomatic children with Down syndrome: cost-effectiveness of preventing lymphoma. Pediatrics. 2006;118(2):594-602.

13. Steingass KJ, Chicoine B, Mcguire D, Roizen NJ. Developmental disabilities grown up: Down syndrome. J Dev Behav Pediatr. 2011;32(7):548-58. 14. Borland LM, Colligan J, Brandom BW. Frequency of anesthesia-related complications in children with Down syndrome under general anesthesia for noncardiac procedures. Paediatr Anaesth. 2004;14(9):733-8. 15. Dickson BC, Streutker CJ, Chetty, R. Coeliac disease: an update for pathologists. J Clin Pathol. 2006;59(10):1008–16. 16. Book L, Hart A, Black J, Feolo M, Zone JJ, Neuhausen SL. Prevalence and clinical characteristics of celiac disease in Downs syndrome in a US study. Am J Med Genet. 2001;98(1):70-4. 17. Charlot L, Abend S, Ravin P, Mastis K, Hunt A, Deutsch C. Nonpsychiatric health problems among psychiatric inpatients with intellectual disabilities. J Intellect Disabil Res. 2011;55(2):199-209. 18. U.S. Department of Agriculture and U.S. Department of Health and Human Services. Dietary guidelines for Americans 2010. http://www. health.gov/dietaryguidelines/dga2010/DietaryGuidelines2010.pdf. Accessed August 11, 2013.

22. Presson AP, Partyka G, Jensen KM et al. Current estimate of Down syndrome population prevalence in the United States. J Pediatr. 2013;163(4):1163-8. 23. New Choice Health (2013). Esophagogastroduodenoscopy cost and procedure information. Accessed Aug 13, 2013 from h t t p : / / w w w. n e w c h o i c e h e a l t h . c o m / D i r e c t o r y / P r o c e d u r e / 1 2 6 / Esophagogastroduodenoscopy. 24. Joda H, Beni V, Alakulppi N et al. Medium-high resolution electrochemical genotyping of HLA-DQ2/DQ8 for detection of predisposition to coeliac disease. Anal Bioanal Chem. 2014;406(12): 2757-69. 25. Wroblova K, Kolorz M, Pav I, et al. Frequencies of HLA-DQ2 and HLADQ8 haplotypes in Czech and Slovak coeliac patients and the healthy population. Acta Biochim Pol. 2014;61(1):191-3. 26. Dalton AJ, McVilly KR. Ethics guidelines for international multicenter research involving people with intellectual disabilities. J Policy Pract Intellect Disabil. 2004;1(2):57-70.

19. Van Gameren-Oosterom HB, Fekkes M, Reijneveld SA et al. Practical and social skills of 16-19-year-olds with Down syndrome: Independence still far away. Res Dev Disabil. 2013;34(12):4599-607.

RESPONDING TO THE NEEDS OF CAREER EM PHYSICIANS

The American Board of Physician Specialties®, through the Board of Certification in Emergency Medicine (BCEM), offers a way for primary care trained physicians with substantial training and experience in Emergency Medicine to demonstrate their mastery of Emergency Medicine. BCEM is one of the nation’s largest EM boards of certification and is the only EM certifying body to recognize PC trained physicians with substantial ER experience. Learn more at: www.abpsus.org/em-pcto

The Utility of Anti-tissue Transglutaminase Antibody-IgA (tTG-IgA) . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

Comparison of Actual and Predicted Emergency Department Wait Times LouAnn H. Woodward, MD Jason Zimmerman, BSN Kristen C. Isom, RN Richard L. Summers, MD

Abstract Objective: Patient dissatisfaction and elopements from overcrowded emergency department (ED) waiting rooms are becoming increasingly common. Previous studies suggest that patients leaving an ED before being seen by a physician presents a legitimate safety concern. Predicted wait times are often communicated to patients in ED waiting rooms as a customer service function and in an attempt to reduce elopements. Common methodologies used by the restaurant industry have been adopted to make these wait predictions. In this study, we compare these predicted wait times to actual wait times experienced by patients. Methods: The actual wait times experienced by patients presenting to the ED of a large academic hospital were determined for a two-week period. These actual times were correlated with the times predicted by a formula typically used to estimate restaurant seating wait times. This wait time was determined as the longest time for a patient already in the waiting room at the moment of the arrival of the new patient to the ED. Pearson’s correlation coefficient (r) was calculated in the comparison analysis. Results: A total of 2051 patient visits were analyzed. Only 33% of the predicted times were within ± 30 minutes of the actual time and 66% of the predicted times underestimated the actual wait times. The correlation between the actual and predicted wait times was r = 0.18. Though none of these patients were informed of their predicted wait time, a total of 4.6% of the patients left before being seen by a physician.

Conclusions: Providing patients in ED waiting rooms with an estimated wait time by means commonly used by the restaurant industry has been shown to significantly decrease the rate of elopements. In this study, we found a poor correlation between the times estimated by this methodology and the actual wait times experienced by patients. In fact, only about a third of the patients received somewhat accurate estimations of their potential wait. These findings suggest that the dynamics of triage and prioritizations in a busy ED are complex and may require a more sophisticated formula for the prediction of wait times. While elopements may diminish when patients are provided with estimated wait times, it is uncertain what impact this practice would have on patient satisfaction.

Introduction Hospitals have become increasingly aware of the importance of patient satisfaction to financial success and often model themselves after other consumer-driven industries. As a result, hospitals have adopted some of the same service strategies these businesses use to cater to their customers. One such strategy that has become progressively more common is the practice of providing emergency department (ED) patients with prospective wait times before being seen by a physician.1 In fact, some hospitals advertise these wait times on billboards, websites, or even cell phone apps.2,3,4 The most commonly employed methodology for providing these predictions of ED wait times is a technique frequently used by restaurants in their estimation of how long it will take to seat guests who arrive without a reservation.5 With this technique, the hostess is able to inform the

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customers of their expected wait times. As consumers, we have become accustomed to this service and it often influences our decision making. Restaurateurs know that if they are unable to tell guests how long their prospective wait will be, they will probably leave. Likewise, a declaration of a specific expectant waiting period also prevents repeated questioning by frustrated guests about the status of their service. Elopements from ED waiting rooms are commonplace and are considered both a customer service and a liability problem for hospitals.6,7,8,9 Therefore, any methodology that can inform patients about the length of time they are expected to wait before seeing a physician could be valuable. However, emergency care is a complex process and the triage procedure is not based on a first come, first served algorithm such as with other service industries. If predicted wait times are wildly erroneous, this could lead to greater frustration and dissatisfaction among patients. In this study, we compared actual ED wait times to those times predicted for patients presenting to a large academic medical center.

between the actual and predicted wait times was r = 0.18 (Figure 2). Though none of these patients were informed of their predicted wait time, a total of 4.6% of the patients left before being seen by a physician. Figure 1: This bar graph represents the numbers of patients (frequency) in blocks of 30 minute time increments that waited to be seen either longer or less than the estimated time.

Methods In a retrospective analysis, the actual wait times experienced by patients presenting to the adult ED of an inner-city academic hospital and Level 1 trauma center with a yearly volume of around 70,000 patient visits were determined for a two-week period. The times of arrival to the ED and times the patient were brought into a room were captured in an electronic tracking system. Excluded from the analysis were patients arriving by ambulance or helicopter and those brought immediately (< 10 minutes) from triage to a room because of the severity of their illness. The actual wait time for each individual patient was inserted in a database that also contained the wait time that would be predicted by a formula typically used to estimate restaurant seating times.5 This wait time was determined as the longest time for a patient already in the waiting room at the moment of the arrival of the new patient to the ED. A correlation comparison of the actual and predicted wait time was performed using Pearson’s correlation coefficient (r). The patients were also divided into groups according to their deviation from the predicted time in ±30 minute increments. The percent of patients for which the prediction was considered relatively accurate (within ±30 minutes of the predicted time) was calculated. The investigational protocol for this study was approved by the Institutional Review Board of the University of Mississippi Medical Center.

Results Data extracted from the tracking system meeting our criteria resulted in a total of 2051 patient visits for analysis. From this database, only 33% of the predicted times were within ± 30 minutes of the actual time and 66% of the predicted times underestimated the actual wait times (Figure 1). The correlation

Figure 2: This graph compares the actual to estimated wait times for patients presenting to the ED to be seen. The Pearson’s correlation coefficient for this comparison is r = 0.18 Correlation Between Predicted and Actual Times

Conclusions Providing patients in ED waiting rooms with an estimated wait time has been shown to significantly decrease the rate of elopements and is thought to be an important customer service initiative.6,10 In this study, we found a very poor correlation between the times estimated by the commonly used methodology and the actual wait times experienced by patients. In fact, only a third of the patients received estimations that were within 30 minutes of their actual times.

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From Figure 1, it appears that patients’ wait times are more likely to be underestimated than overestimated. If patients are given wildly erroneous information regarding their expected wait, it is uncertain what impact this might have on their satisfaction with their ED experience. There is some evidence that underestimating wait predictions adversely effects patient satisfaction.11 However, such a unilateral strategy might also result in a larger number of people leaving the ED without being seen by a physician because of the cited wait. Intuitively, most people would expect that underestimations of wait times would result in significant patient anguish over the delays and create animosity toward triage personnel. Despite the potential problems associated with misrepresentations of wait times, many hospitals continue to pursue this idea. Perhaps just as the restaurant industry has tried to refine their wait estimations, hospitals could adopt more sophisticated methods that could provide for more accurate predictions. Modern digital technologies could be used in conjunction with complex algorithms that include patient acuity and flow calculations along with staffing inputs to provide better information concerning expected ED wait times.12 However, developing such methods is more difficult for health care systems as compared to other service industries and would have to be routinely validated for accuracy. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

LouAnn H. Woodward, MD, is professor of emergency medicine at the University of Mississippi Medical Center in Jackson, MS.

References 1.

Rae R, Wilson A, Higgins A, Brown J, Labriola J, O’Neill B. Setting patient expectations of waiting times improves patient satisfaction in the emergency department[abstract]. Acad Emerg Med. 2008;15(Suppl 1):S110.

Emergency departments post wait times on the web. ED Manag. 2010;22(1):10-1.

Patients can text EDs for wait times. ED Manag. 2010;22(6):64-5.

Xie B, Youash S. The effects of publishing emergency department wait time on patient utilization patterns in a community with two emergency department sites: a retrospective, quasi-experiment design. Int J Emerg Med. 2011;4(1):29.

Handel DA, Fu R, Daya M, York J, Larson E, John McConnell K. The use of scripting at triage and its impact on elopements. Acad Emerg Med. 2010;17(5):495-500.

Guttmann A, Schull MJ, Vermeulen MJ, Stukel TA. Association between waiting times and short term mortality and hospital admission after departure from emergency department: population based cohort study from Ontario, Canada. BMJ. 2011;342:d2983

Bambi S, Scarlini D, Becattini G, Alocci P, Ruggeri M. Characteristics of patients who leave the ED triage area without being seen by a doctor: a descriptive study in an urban level II Italian University Hospital. J Emerg Nurs. 2011;37:334-40.

Will longer wait times mean more ED lawsuits? ED Manag. 2010;22(5):57-8.

Naumann S, Miles JA. Managing waiting patients’ perceptions: the role of process control. J Manag Med. 2001;15(4-5):376-86.

10. Arendt KW, Sadosty AT, Weaver AL, Brent CR, Boie ET. The leftwithout-being-seen patients: what would keep them from leaving? Ann Emerg Med. 2003;42(3):317-23. 11. Rae R, Wilson AG, Brown J, Higgins A, O’Neil BJ. Does providing patients with overestimates of anticipated waiting times improve perception of wait times in the emergency department? Ann Emerg Med. 2008;52:S111. 12. Leet L, Taylor B. Method for accurately quoting wait time for a restaurant table. QSR Automations, Inc. Patent No. US-74,756,745-B2. 2010.

Jason Zimmerman, BSN, is director of adult emergency department at the University of Mississippi Medical Center in Jackson, MS. Kristen C. Isom, RN, is a stroke nurse administrator at Anderson Regional Medical Center in Meridian, MS. Richard L. Summers, MD, is professor of emergency medicine at the University of Mississippi Medical Center in Jackson, MS.

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Factors Affecting Rural Patients’ Compliance with Screening Colonoscopy: Importance of Distance and Physician Arsheeya Mashaw, MD Justin T. Arcury, MS

Abstract Introduction: Rural populations face unique barriers to obtaining colorectal cancer (CRC) screening. This study assessed factors associated with rural patients obtaining a colonoscopy, including distance to services, patient knowledge, and discussion with a physician. Methods: Self-administered questionnaires were completed by 106 hospital patients scheduled for an outpatient colonoscopy at a rural hospital and 100 general patients in a rural primary care practice. These questionnaires assessed patient characteristics and opinions regarding factors affecting the choice of where to get a colonoscopy. Univariate analyses delineated the importance of distance and other factors, such as knowledge about a colonoscopy, discussion with health care provider, and referral by provider. Results: Most (95.3%) of hospital patients obtaining a colonoscopy had received information about the procedure from their physician, and two-thirds (66.3%) of general clinic patients had discussions about colonoscopy with their physician. The majority of patients obtaining a colonoscopy (68%) stated they would not have gotten the procedure if it was not near their home. Subjects stated that convenient location was one of the most important factors influencing their decision to obtain a colonoscopy. Being referred by their physician and physician reputation were important whether obtaining a colonoscopy near or far from home.

Conclusions: Travel time (distance) played a significant role in rural patients’ decisions to obtain a colonoscopy. Patients having to travel greater distances were less likely to undergo colonoscopy than those traveling shorter distances.

Introduction Colorectal cancer (CRC) is the third most common cause of cancer death among men and women in the United States. There is a lifetime risk of 5.17% for US men and 4.78% for US women.1 In 2009, 136,717 people in the United States were diagnosed with CRC, including 70,223 men and 66,494 women. In the same year, 51,848 people in the United States died from CRC, including 26,806 men and 25,042 women.2 Screening can reduce both the incidence of new cases and deaths from CRC.3-12 Screening can detect precancerous polyps which can be removed; precancerous polyps have a high rate of transforming into CRC. People found with localized CRC have a 5-year survival rate of about 90% compared to 66% for people diagnosed with regional involvement and only 9% for people diagnosed with distant metastases.13 Screening for CRC requires an ongoing program of appropriate testing and follow-up. Recommendations for CRC screening take into account several factors, including effectiveness, sensitivity, false-positive rate, safety, cost, and patient preference. Three major US guidelines (American Cancer Society, American Gastroenterological Association, and US Preventive Ser-

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vices Task Force) have similar recommendations, with screening modalities including fecal occult blood testing (FOBT) annually, sigmoidoscopy and barium enema every 5 years, or colonoscopy every 10 years. However, despite the multitude of screening options, CRC screening adherence remains suboptimal. Rates for CRC screening in the United States for 2010 were 58.6%.15 Screening rates are higher among adults who are insured, better educated, non-Hispanic, or have a usual source of medical care.14 Rural residents have lower rates of appropriate CRC screening compared to their urban counterparts.16 Since rural populations lag in colonoscopy or sigmoidoscopy use as compared to their urban counterparts,17 rural communities may be at higher risk of CRC due to lower screening rates.18 Barriers that rural adults face which limit CRC screening include inability to pay for the procedure, inadequate health insurance, low educational attainment, and a limited number of endoscopists.19,20 Young et al suggest that visiting a doctor within the last year and counseling by a doctor also contribute to having appropriate CRC screening.21 Greiner et al suggest that CRC screening among rural primary care patients is related to adequacy of physician CRC screening discussions but not access or availability to endoscopic procedures.22 Little is known about CRC screening in rural communities where physical distance from health care services is often a barrier. However, Arcury et al point out that distance is a factor in the utilization of health services among rural residents.23 Many rural communities lack the facilities, the endoscopists, or the staff to provide CRC services. Newman et al have shown that properly trained family physicians are capable of performing colonoscopies safely and competently; however, only 5% of family physicians offer colonoscopy services.24 The goal of this study was to delineate factors associated with rural patients’ decisions to get a colonoscopy for CRC screening. Specifically, it considers distance to services, as well as patient knowledge, discussion with health provider, and referral by physician.

Methods Study Setting This study was conducted in Page County, Virginia. This region is secluded in the Shenandoah Mountains of West Central Virginia. It is bordered on the west by the Shenandoah National Park and on the east by the Massanutten Mountain Range. It has a limited road system, high rates of poverty, and poor health.25 According to the US Department of Health and Human Services, a region with an Index of Medical Underservice score of 62 or less is considered a medically underserved area. Page County is a medically underserved area with an Index of Medical Underservice score of 52.26 The county had an estimated population of 23,895 in 2012, and the city of Luray, the county seat, had a population of 4,878.27 The county has one hospital, Page Memorial Hospital, which is designated as a Critical Access Hospital by the Centers for Medicare and Medicaid Services.28

Participants Two patient samples were recruited to this study: hospital ambulatory surgery patients scheduled for a screening colonoscopy and general clinic patients. Patients attending an ambulatory surgery setting at Page Memorial Hospital between January 2012 and December 2012 were asked to participate. Patients who were ages 50 to 75 years (the generally accepted age range for CRC screening with a colonoscopy) were provided a one-page, anonymous questionnaire to complete in the waiting area prior to their screening colonoscopy. A total of 106 hospital patients were recruited. Second, patients in a Luray outpatient clinic between April 2012 and December 2012 were asked to participate. Patients were invited to complete a one-page questionnaire if they were 50 to 75 years old; one 48-year-old patient was included because of a family history of CRC. A total of 100 clinic patients were recruited. No hospital or clinic patients refused participation. The research protocol was reviewed and approved by the Valley Health Institutional Review Board. Valley Health is the parent hospital system of Page Memorial Hospital.

Data Collection Clinic and hospital participants completed self-administered surveys. Each survey included items addressing personal and health characteristics and factors affecting their choice of a hospital for a colonoscopy.

Measures Patient personal and health characteristics included gender, age (44-54, 55-59, 60-64, 65-69, 70-79), annual family income (<$30,000, $30,000 to $50,000, >$50,000), education (<high school, high school, >high school), health insurance, and having seen a primary care provider in the past 12 months (Table 1). Knowledge about colonoscopy included whether the hospital and clinic patients reported being up to date on CRC screening. Hospital patients also indicated whether they understood why they were getting a colonoscopy and from whom they received information about the procedure (discussion with physician, self-study, or other). Clinic patients indicated whether they knew what a colonoscopy was and if their health care provider had spoken to them about a colonoscopy. Hospital patients reported on factors related to where they wanted to get a colonoscopy, including whether they would have gotten the procedure if it were not available near home, how far in minutes they drove that day to get the procedure (0-10, 11-15, 16 or more minutes), distance in minutes they would be willing to drive for a colonoscopy (30, 45, or 60-90 minutes), and whether they were more likely to get the procedure because it was close to their home (strongly agree, agree, disagree/distance did not matter). Hospital patients also selected the three top factors that influenced their decision to have a colonoscopy at Page Memorial Hospital from a list that included convenient location, physician reputation, physician referral, staff reputation, and flexible scheduling.

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Clinic patients selected the three top factors that would influence whether they might have a colonoscopy at three different types of hospitals. The possible factors included convenient location, physician reputation, physician referral, staff reputation, flexible scheduling. The three types of hospitals were local (Page Memorial Hospital), regional (Harrisonburg or Winchester Hospitals), and statewide (University of Virginia Hospital, Charlottesville).

Table 1: Personal and Health Characteristics for Hospital and Clinic Samples

Personal and Health Characteristics

Hospital

Clinic

Gender

Analysis

Female

54.5

55.0

Frequency counts and percentages were computed for personal and health characteristics (Table 1), knowledge about colonoscopies for hospital and clinic samples, factors related to where participants want to get colonoscopies in the hospital sample (Table 2), and factors important to both hospital and clinic patients in getting a colonoscopy (Table 3). Frequency counts and percentages were also computed for cross tabulations between variables in Tables 1 through 3 and between indicators of propinquity importance for hospital patients in getting a colonoscopy and personal and health characteristics. Univariate analyses associated with each cross tabulation were conducted using chi-squared or Fisher’s exact test where appropriate. Multiple testing was adjusted for using a Bonferroni correction, and a resulting significance level of 0.0001 was used for all tests. All analyses were

Male

45.5

45.0

44-54

25.7

18.0

55-59

21.0

28.0

60-64

12.4

15.0

65-69

20.0

23.0

70-79

21.0

16.0

< $30,000

50.0

$30,000 to $50,000

30.4

25.6

>$50,000

19.6

24.4

<High school

17.9

High school

40.6

>High school

41.5

10.1

Yes

89.9

conducted using SAS 9.1 (SAS Institute, Cary, NC).

Results Personal and Health Characteristics Hospital and clinic samples possessed similar personal characteristics (Table 1). About 55% of both samples were female, and they included the same distributions of ages. They were also similar in annual family income with half having annual family incomes of less than $30,000 and about 20% having family incomes of more than $50,000. Most (82.1%) of the hospital participants had a high school education or greater. Most (89.9%) of the clinic participants had health insurance. Almost all hospital and clinic participants had seen a primary care provider in the past 12 months. Hospital and clinic patients differed somewhat in their knowledge of colonoscopy screening. Although most (72.9%) of the hospital patients were up to date with CRC screening, fewer than half (48.5%) of the clinic patients were up to date. Almost all (99.0%) of the hospital patients noted that they knew why they were getting a colonoscopy, and almost all (94.0%) of the clinic patients stated that they knew what a colonoscopy was. Almost all of the hospital patients (95.3%) had received information about the procedure through discussion with their physician, but only 66.3% of the clinic patients noted that their health care provider had spoken with them about a colonoscopy.

Age (years)

Annual Family Income

Education

Health Insurance

Seen Primary Care Provider in Past 12 Months No

2.1

5.2

Yes

97.9

94.8

Factors Affecting Where Patients Want to Have Colonoscopies Travel time (distance to services), and location were extremely important to hospital patients when getting a colonoscopy (Table 2). Most (68.0%) stated they would not have gotten the procedure if it was not near to their home. Most noted that they

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had driven 15 or fewer minutes to get the procedure, with 33.0% having driven 0 to 10 minutes and 43.6% having driven 11 to 15 minutes. Most (65.4%) indicated that they would be willing to travel 30 minutes to get a colonoscopy. Only 16.4% indicated that they would be willing to travel 45 minutes and 18.3% indicated that they would be willing to travel 60 or 90 minutes. Finally, most strongly agreed (46.2%) or agreed (39.6%) that they were more likely to get the test because it was close to home. The importance of these factors did not have statistically significant differences by any of the personal and health or colonoscopy knowledge characteristics. However, stating that they would not get the procedure if it were not near home was significantly correlated with stating that they would be willing to drive 30 minutes to get a colonoscopy (84.1% agreement, p < 0.0001), and with strongly agreeing (58.6%) and agreeing (35.7%) that they were more likely to get the test because it was close to home (p < 0.0001). Table 2: Factors Related to Where Participants Want to Get Colonoscopies: Hospital Sample

Factors Related to Getting a Colonoscopy

Gotten Procedure If It Was Not Near Home No

68.0

Yes

32.0

How Far Did You Drive Today (Minutes) 0-10

33.0

11-15

43.6

16 or more

23.4

important to few patients (6.6% for both) in getting a colonoscopy at Page Memorial Hospital. The importance of these factors did not have statistically significant differences by any of the personal and health characteristics, colonoscopy knowledge characteristics, or most factors related to where patients want to get colonoscopies. Most of the clinic patients (89.0%) selected convenient location as one of the three top factors that might influence their decision to have a colonoscopy at Page Memorial Hospital (Table 3). Other important factors listed among clinic patients in whether they would get a colonoscopy at Page Memorial Hospital were physician reputation, selected by 83.0% of patients, and physician referral, selected by 69.0% of patients. Staff reputation (27.0%) and flexible scheduling (32.0%) were also selected by sizable numbers of clinic patients as important in a decision to get a colonoscopy at Page Memorial Hospital. Table 3: Factors Important in Deciding Where to get a Colonoscopy among Clinic Participants for Local, Regional and Statewide Hospitals

Colonoscopy Locations and Factors

Convenient Location

89.0

Physician Reputation

83.0

Physician Referral

69.0

Staff Reputation

27.0

Flexible Scheduling

32.0

Local: Page Memorial Hospital

Regional: Harrisonburg or Winchester Hospitals

Distance (Minutes) Willing to Drive to Get a Colonoscopy 30

65.4

16.4

60 or 90

18.3

More likely to get test because it was close to my home Strongly agree

46.2

Agree

39.6

Disagree/ distance didn’t matter

14.1

Most of the hospital patients (84.9%) selected convenient location as one of the three top factors important to whether they would get a colonoscopy at Page Memorial Hospital. Also important were physician reputation (63.2%), and physician referral (57.6%). Staff reputation and flexible scheduling were

Convenient Location

28.6

Physician Reputation

85.7

Physician Referral

82.7

Staff Reputations

49.0

Flexible Scheduling

43.9

Statewide: University of Virginia Hospital, Charlottesville Convenient Location

15.6

Physician Reputation

81.3

Physician Referral

77.1

Staff Reputation

60.4

Flexible Scheduling

40.6

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Convenient location became much less important when clinic patients noted the three top factors important to whether they would get a colonoscopy at a regional hospital (Harrisonburg or Winchester) (28.6%) or statewide hospital (University of Virginia, Charlottesville) (15.6%). Physician reputation maintained its importance for whether a subject would get a colonoscopy at a regional (85.7%) or statewide hospital (81.3%), and physician referral gained in importance (82.7% and 77.1%, respectively, compared to 69.0% in selecting Page Memorial Hospital). More clinic patients selected staff reputation (49.0% and 60.0%, respectively, compared to 27.0% in selecting Page) and flexible scheduling (43.9 and 40.6%, respectively, compared to 32.0% in selecting Page) as an important factor in selecting the regional or statewide hospitals. The importance of these factors did not have statistically significant differences by any of the personal and health characteristics, colonoscopy knowledge characteristics, or most factors related to where patients want to get colonoscopies.

Discussion This study relates distance to services (travel time) to rural patients’ likelihood of obtaining colonoscopies for CRC screening. This study demonstrates the positive influence that patient knowledge about colonoscopies and physician-patient discussions about colonoscopies have in influencing rural patient behavior when deciding to get appropriate CRC screening. Little research addresses the importance of service availability and distance for rural residents in getting appropriate CRC screening. Arcury et al indicate the general importance of distance to care in rural communities.23 Simpson et al report that improving access to health care at the county and individual levels through expanded health insurance coverage could improve CRC screening, but they do not address the effects of distance on access.29 One study that considered distance to services and obtaining a colonoscopy for rural populations found no significant correlation between distance and service utilization; however, it did not discern actual differences in geographic distance to services or travel time to services.19 This analysis indicates that patients in rural hospital and clinic settings consistently considered convenient location an important factor in deciding to have a colonoscopy. They were much more likely to get a colonoscopy if it were available within a 30 minute travel time. Many subjects, when asked in the hospital setting, said that they would not have gotten a colonoscopy if it were not available nearby. Location and availability play a pivotal role in patients obtaining a colonoscopy; having this procedure available in rural areas will increase the rates of CRC screening. However, providing procedures like colonoscopies in rural areas is always a challenge. As described earlier, few family physicians are adequately trained to offer colonoscopy services. However, they are capable of performing colonoscopies safely and competently compared to their specialist counterparts.24 Ways to increase availability would include continued training of endoscopic skills for primary care physicians in rural areas.

Previous studies have shown that physician reputation and referral by a physician were also important to patients getting a colonoscopy.30,31 Similarly, this study suggests that physician reputation and being referred by one’s physician were rated as important factors in getting a colonoscopy. Previous studies have shown how important physician discussions with patients are to patients obtaining colonoscopies.19,20,32 Almost all of the patients in the hospital setting had received information about a colonoscopy from their physician, but only 66.3% of the clinic patients noted that their health care provider had spoken with them about a colonoscopy. Another finding shows that the patient population getting the procedure at the local hospital had the highest percentage of patients considering convenient location (travel time) as more important (89%). Thus they were less likely to travel to the regional and university hospital. Those who chose to go to the regional and university hospitals placed more importance on physician reputation than convenient location (travel time) in their decision to go for colonoscopy. In developing policies or public education materials, these differences should be taken into consideration. This study has limitations. It was a cross-sectional study and the major outcome variables were determined by self-report. However, prior validation studies of self-reported CRC screening have shown good correspondence to chart and billing record audits.33,4 The study was conducted in a single rural county, and it utilized a systematic, but not a randomized, sampling technique. Therefore, generalizations of the results must be made with caution. These results indicate that patients consider location and distance when assessing if or where they will have a colonoscopy. They also show the importance of provider referral and provider reputation. Comparative studies are needed to compare CRC screening rates in rural communities in which colonoscopies are available to rates in other rural communities where they are not. This research should investigate the importance of patient-physician discussion and physician provided education in patients’ decisions to have a colonoscopy in these communities.

Acknowledgement The Program in Community Engagement, Wake Forest Translational Science Institute provided suggestions and comments on the design of the research and the formulation of this manuscript. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Arsheeya Mashaw, MD, is a family physician and geriatrician at Page Memorial Hospital in Luray, VA. Justin T. Arcury, MS, is a statistical consultant and graduate student at Wake Forest University in Winston-Salem, NC.

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References 1.

Siegel R, Naishadham D, Jemal A. Cancer statistics, 2013. CA Cancer J Clin. 2013;63(1):11-30.

US Cancer Statistics Working Group. United States Cancer Statistics: 1999-2009 Incidence and Mortality Web based report. Atlanta, GA: Department of Health and Human Services, Centers for Disease Control and Prevention; 2013. http://www.cdc.gov/cancer/colorectal/statistics/. Accessed May 17, 2013.

Winawer SJ, Fletcher RH, Miller L, et al. Colorectal cancer screening: clinical guidelines and rationale. Gastroenterology. 1997;112(2):594-642.

Mandel JS, Bond JH, Church TR, et al. Reducing mortality from colorectal cancer by screening for fecal occult blood. Minnesota colon cancer control study. N Engl J Med. 1993;328(19):1365 -71.

18. Kinney AY, Harrell J, Slattery M, Martin C, Sandler RS. Rural-urban differences in colon cancer risk in blacks and whites: the North Carolina Colon Cancer Study. J Rural Health. 2006; 22(2):124-30. 19. Rowland D, Lyons B. Triple jeopardy: rural, poor, and uninsured. Health Serv Res. 1989;23(6):975-1004. 20. Conger RD. The special nature of Rural America. In: Robertson EB, Sloboda Z, Boyd GM, Beatty L, Kozel NJ, eds. Rural Substance abuse: State of Knowledge and Issues. NIH Publication, Vol.97-4177. Rockville, MD: US Department of Health and Human Services, NIH; 1997.p.37-52. 21. Young WF, McGloin J, Zittleman L, West DR, Westfall JM. Predictors of colorectal screening in rural Colorado: testing to prevent colon cancer in the high plains research network. J Rural Health. 2007; 23(3):238-45. 22. Greiner KA, Engelman KK, Hall MA, Ellerbeck EF. Barriers to colorectal cancer screening in rural primary care. Prev Med. 2004;38(3):269-75.

Mandel JS, Church TR, Ederer F, Bond JH. Colorectal cancer mortality: effectiveness of biennial screening for fecal occult blood. J Natl Cancer Inst. 1999;91(5):434-7.

23. Arcury TA, Preisser JS, Jr., Gesler WM, Powers JM. Access to transportation and health care utilization in a rural region. J Rural Health. 2005;21(1):31-8.

Mandel JS, Church TR, Bond JH et al. The effect of fecal occultblood screening on the incidence of colorectal cancer. N Engl J Med. 2000;343(22):1603-7.

24. Newman RJ, Nichols DB, Cummings DM. Outpatient colonoscopy by rural family physicians. Ann Fam Med. 2005;3(2):122-5.

Selby JV, Friedman GD, Quesenberry CP, Weiss NS. A case-control study of screening sigmoidoscopy and mortality from colorectal cancer. N Engl J Med. 1992;326(10):653-7.

25. Cockley, David R. Page County, Health Needs Assessment, Health Resource Inventory, March 2001;6-7. Available at: http://www.iihhs.jmu. edu/healthplace/pdfs/page_health_assessment_part1.pdf. Accessed Jan 13, 2013.

Atkin WS, Hart A, Edwards R, et al. Uptake, yield of neoplasia, and adverse effects of flexible sigmoidoscopy screening. Gut. 1998;42(4):5605.

26. Health Resources and Services Administration, Guidelines for Medically Underserved Areas. Available at: http://www.hrsa.gov/shortage/mua/ index.html. Accessed February 5, 2013.

Newcomb PA, Norfleet RG, Storer BE, Surawics TS, Marcus PM. Screening sigmoidoscopy and colorectal cancer mortality. J Natl Cancer Inst. 1992;84(20):1572-5.

27. United States Census Bureau, 2010 Census Data, Page County Virginia. Available at: http://quickfacts.census.gov/qfd/states/51/51139.html. Accessed May 17, 2013.

10. Winawer SJ, Zauber AG. Colonoscopic polypectomy and the incidence of colorectal cancer. Gut. 2001;48(6):753-4. 11. Winawer SJ, Zauber AG, Ho MN, et al. Prevention of colorectal cancer by colonoscopic polypectomy. The National Polyp Study Workgroup. N Engl J Med. 1993;329(27):1977-81.

28. Department of Health and Human Services, Centers for Medicare and Medicaid Services. Critical Access Hospital Designation.Available at:http://www.cms.gov/Outreach-and-Education/Medicare-LearningNetwork-MLN/MLNProducts/downloads/critaccesshospfctsht.pdf. Accessed May 17, 2013.

12. Muller AD, Sonnenberg A. Prevention of colorectal cancer by flexible endoscopy and polypectomy. A case-control study of 32,702 veterans. Ann Intern Med. 1995;123(12):904-10.

29. Stimpson JP, Pagán JA, Chen LW. Reducing racial and ethnic disparities in colorectal cancer screening is likely to require more than access to care. Health Aff (Millwood). 2012;31(12):2747-54.

13. Greenlee RT, Hill-Harmon MB, Murray T, Thun M. Cancer statistics, 2001. CA Cancer J Clin. 2001;51(1):15-36.

30. Beydoun HA, Beydoun MA. Predictors of colorectal cancer screening behaviors among average-risk older adults in the United States. Cancer Causes Control. 2008;19(4):339-59.

14. Meissner HI, Breen N, Klabunde CN, Vernon SW. Patterns of colorectal cancer screening uptake among men and women in the United States. Cancer Epidemiol Biomarkers Prev. 2006; 15(2):389-94. 15. Centers for Disease Control and Prevention (CDC). Cancer screening United States, 2010. MMWR Morb Mortal Wkly Rep. 2012;61(3):41-5. 16. Coughlin SS, Thompson TD. Colorectal cancer screening practices among men and women in rural and nonrural areas of the United States, 1999. J Rural Health. 2004; 20(2):118-124. 17. Cole, AM, Jackson JE, Doescher M. Urban–rural disparities in colorectal cancer screening: cross-sectional analysis of 1998–2005 data from the Centers for Disease Control’s Behavioral Risk Factor Surveillance Study. Cancer Med. 2012;1(3):350-6. http://onlinelibrary.wiley.com/ doi/10.1002/cam4.40/full. Accessed April 15, 2013.

31. Christie J, Hooper C, Redd WH, et al. Predictors of endoscopy in minority women. J Natl Med Assoc. 2005;97(10):1361-8. 32. Benarroch-Gampel J, Sheffield KM, Lin YL, Kuo YF, Goodwin JS, Riall TS. Colonoscopist and primary care physician supply and disparities in colorectal cancer screening. Health Serv Res. 2012;47(3 Pt 1):1137-57. 33. Madlensky L, McLaughlin J, Goel V. A comparison of self-reported colorectal cancer screening with medical records. Cancer Epidemiol Biomarkers Prev. 2003;12(7):656–9. 34. Baier M, Calonge N, Cutter G, et al. Validity of self-reported colorectal cancer screening behavior. Cancer Epidemiol Biomarkers Prev. 2000;9(2):229 –32.

Factors Affecting Rural Patients’ Compliance . . .

American Journal of Clinical MedicineÂŽ â&#x20AC;˘ Summer 2014 â&#x20AC;˘ Volume Ten Number Two

The Declining Number of Family Physicians Practicing Obstetrics: Rural Impact, Reasons, Recommendations and Considerations Descriptive Research Daniel M. Avery, Jr, MD John T. McDonald, Jr, MD, CPA

Summary Maternity care in the United States is in jeopardy. There is a need for physicians trained in obstetrics, including preconception, prenatal, delivery, and postpartum care. Almost 50% of counties in the United States have no obstetrical provider.1 The importance and high quality of family medicine obstetrics has been documented repeatedly2-7, but the number of family physicians providing maternity care in the United States continues to decline.8-12 This is also an international trend as reported in Australia and Canada.3,13 Since family medicine residencies began in 1972, family physicians have been trained to provide obstetrical care, but it was not until the accreditation changes of 1995 that residencies required family practice faculty to participate in this training.14 This requirement revealed that many faculty of the family medicine programs did not teach or believe in the reality of obstetrics by family physicians.15 This led to a backlash from some family practice residencies who claimed hospital and community circumstances made teaching delivery skills unnecessary, unaffordable, or politically impossible. This led to a three-tiered proposal for obstetrics skills within family medicine.16 Tier 3 came to be known as surgical family medicine obstetrics because it included cesarean section training for family physicians.17-22 Collectively, these skills remain vital to the health of communities here and in the developing world. More and more family physicians in the United States have stopped delivering babies in rural, underserved areas where family physicians may be the only obstetrical providers.23-25 With the trend of increasing

shortages of both obstetrician-gynecologists (OB/GYNs) and family physicians that provide maternity care, rural areas and underserved communities are at the greatest risk for losing maternity care services.26,27 Family medicine with obstetrics has emerged as a cost effective solution offering improvements in access, cost, and quality.2-7, 28,29 For example, in states with the lowest cesarean section rates, almost half of the family physicians deliver babies.21 This paper describes reasons physicians stop providing obstetrical care, and this paper makes recommendations for the growth of family medicine with obstetrics.

Background Fewer medical school graduates are choosing primary care and family medicine, and fewer of those are practicing obstetrics when they enter practice.8 Sometimes the decrease in family physicians providing obstetrical care may be due to a hospital closing its obstetrical services or even a hospital closing entirely.10 A decrease in rural maternity care providers has been associated with poor perinatal outcomes.13 As of 2012, only 10% of family physicians in this country were providing obstetrical care.11 Family medicine is the only residency that is growing in the United States,24 OB/GYN is not.30 The number of obstetrical providers in the United States continues to decline. A recent paper by Rayburn et al stated that almost 50% of counties in the United States have no obstetric provider, which affects approximately ten million women.1 Many of these women live in rural, underserved areas where the need for providers is greatest.1 This deficit increases the

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maternal and neonatal morbidity and mortality risk.1 There is a growing tendency for new OB/GYN residency graduates to congregate in urban areas, which contributes to this maldistribution of providers.1

patients, especially funded pregnant patients, can make providing obstetrical care financially impossible and can make keeping up one’s skills a difficult task.

Although there has been a decrease in OB/GYN residencies from 306 to 246 since 1993, the number of OB/GYN residency graduates has not declined. The remaining programs have grown in size to absorb trainees from programs that were closed.30

Table 1: Reasons Family Physicians Stop Practicing Obstetrics

Fewer medical students are choosing OB/GYN as a career. Those that do choose OB/GYN as a career often choose subspecialty training, not general OB/GYN, and these graduates do not practice in rural, underserved areas.4 There are no current plans to increase residency programs in OB/GYN.30 The current attrition rate of retiring OB/GYN physicians approximately equals the number of new OB/GYN residency graduates.30 When physicians stop delivering babies, they rarely return regardless of the circumstances.9 Hospitals that stop providing obstetric services almost never resume that service. Family physicians are in a unique position allowing them to give up obstetrics and still make a living, unlike most OB/GYNs.

Why Do Family Physicians Stop Practicing Obstetrics? There are many reasons that physicians stop providing obstetrical care. Table 1 lists some of the reasons found in the literature and others collected by one of the authors (DMA). Legal: More physicians stop delivering babies because of malpractice litigation than any other reason.9 The fear of litigation is a legitimate concern of any medical practitioner today; however, it haunts those who practice obstetrics. Most OB/GYN physicians in Alabama report that they have been sued at least once. Even when physicians have done their very best, bad outcomes occur. Regardless of the verdict, physicians leave the courtroom asking themselves, “Is this really worth it?”9 It is not surprising that many obstetrical providers do not encourage their children who are training to be physicians to specialize in obstetrics. The fear of being sued is pervasive. The suggestion or verbal threat of suit, requests for medical records, subpoenas, letters from attorneys, and correspondence from malpractice carriers can be overwhelming.9 Numerous suits or even a single exorbitant plaintiff verdict may be sufficient for a physician to quit providing obstetrical care.9 Certainly, the current legal system has forced a disproportionate financial burden on obstetrical providers that is not commensurate with current financial reimbursement plans. Patients: Patients themselves are a significant part of physicians stopping obstetrics. Patients make unreasonable demands and have unreasonable expectations of their physicians. They may even threaten lawsuits, transfer care to another physician, or register complaints with administration or the hospital if their demands are not met.9 Difficult, demanding, noncompliant, or litigious patients can be trying to even the most patient, well-intentioned physicians.9 Inadequate numbers of pregnant

LEGAL

• • • •

PATIENTS

• Patient demands9 • Difficult patients9 • Patients transferring to another provider • Inadequate numbers of patients

PERSONAL HEALTH

• Physical exhaustion9 • Impairment9 • Debilitating illness9

• • • PSYCHOLOGICAL • • •

Malpractice litigation9 Verbal threat of a suit9 Plaintiff verdict Numerous suits9

Fear of being sued9 Fear of peer review9 Feeling unappreciated Perception of not doing a good job Intimidation by new providers9 Intimidation by new procedures9

PERSONAL LIFE

• Stresses at home19 • Childcare issues19 • Career change19

BAD OUTCOMES

• • • •

Obstetrical disasters Fetal demise Maternal death Permanent birth injury

• • • •

Inability to obtain privileges Closure of hospital19 Closure of obstetrical services Lack of hospital commitment and support

HOSPITALS

GOVERNMENT REGULATIONS

• Electronic medical records • Computerized physician order entry (CPOE)34 • Meaningful use35 • Audits34,35

TECHNICAL

• High cesarean section rate • Elective inductions < 39 weeks • Not keeping up with new advances • Waning technical skills • Inability to obtain new technology • Not appreciating one’s limitations

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Table 1: continued.

FINANCIAL

• • • •

Unaffordable malpractice premiums9 Decreased reimbursement Poor practice management Inadequate financial planning

POLITICAL

• Pressure by OB/GYNs not to deliver1,15,19 • Refusal of hospital to privilege family physicians • Discouragement by hospital from practicing high-risk obstetrics

• Insufficient breadth of exposure during training • Inadequate patient numbers during training • Inadequate patient numbers to keep EDUCATIONAL skills up • Not staying current with the standard of care • Never learned ultrasound • Did not become board certified

ATTRITION

• • • •

Retirement Stopped delivering Moved to another care area Career change

LACK OF PHYSICIAN SUPPORT

• Lack of new recruits19 • Lack of call group support19 • Lack of OB/GYN back-up and referral services • Lack of availability and support from other specialties

Personal Health: Due to increasing age, many family physicians that practice obstetrics and obstetrician-gynecologists choose to end their obstetrics careers. Physicians become physically exhausted and unable to tolerate the long hours and physical demands of staying up all night at a patient’s bedside.9 Time and effort reports at the University of Alabama indicate that some obstetricians work in excess of 120 hours per week.9 Illness or age may preclude the practice of obstetrics. It is rare to see a physician over the age of 60 who continues to provide obstetrical care. Lack of sleep and prolonged bedside care due to worrisome fetal heart rate tracing can contribute to discouragement.9 Discouragement and despair often lead to impairment from a variety of causes.9 Although physicians champion good health with their patients, they seldom take good care of themselves. Psychological: The fear of being sued is all-encompassing. A suit is a humiliating, expensive, belittling, embarrassing, psychologically draining, and demeaning experience.9 Suits can extend over years and progress slowly. Physicians fear peer review for concern that their care may fall under scrutiny and may perceive peer review as insinuating poor care. Discouraged and

tired, physicians that deliver babies often feel unappreciated for their hard work, long hours, and loss of sleep.9 Physicians often wonder what they have done wrong when patients transfer their care to another provider.9 Young, recently trained physicians are a threat to older physicians. The “new kid on the block” always attracts a few long-time patients who want someone new, young, or different.9 Patient allegiance is becoming a thing of the past.31 New procedures can be threatening and intimidating to older physicians, especially if they do not plan to learn the new procedures and patients inquire about them.9 Personal Life: As a solo practitioner, one of the authors (DMA) once was away from home for three continuous weeks. Absence from home leads to marital problems, childcare issues, and often results in career changes including stopping obstetric services to spend more time at home.19 Physicians that are away from home a lot miss important parts of their children growing up, which may lead to resentment and dissatisfaction with practicing obstetrics. Family physicians are trained in many areas and, unlike many other specialties, can do well practicing other areas of family medicine. Today, it is unusual to see obstetrician-gynecologists in private practice give up obstetrics and survive on general gynecology unless they have a special niche. Looking back over their careers, physicians that practice obstetrics often regret their choice of specialty. Personal issues often lead to career changes including discontinuing obstetrics.19 Clearly, work-life balance issues need to be a top priority for the OB/GYN profession as well as those family medicine providers that practice obstetrics. Bad Outcomes: Bad outcomes, regardless of etiology or fault, make physicians question if it is time to stop delivering babies.31-33 Obstetrical disasters, such as ruptured uteri, maternal strokes, and maternal deaths; neonatal disasters, such as an undeliverable shoulder dystocia leading to fetal death; permanent birth injuries, such as cerebral palsy, Erb’s palsy, fractured cervical spine from a breech delivery; and fetal demise often equate with poor care, poor judgment, poor quality, and lawsuits despite the best efforts of the obstetrical provider. Bad outcomes are often the inciting stimulus to end maternity care or “hang it up.” Plaintiffs’ verdicts and exorbitantly large monetary awards often cause significant increases in malpractice insurance premiums that are funded by the obstetrical provider. These increases often make it financially impossible for the physician to continue providing good obstetrical care. Hospitals: Hospitals may not grant family physicians privileges in obstetrics or may grant limited privileges without cesarean section capabilities.15,19 There is the notion that family physicians delivering babies in rural, underserved areas may be appropriate but not so in urban areas. While it may be possible to provide prenatal care locally and then deliver elsewhere or refer patients for delivery, it is often not worth the extra work. Reimbursement for prenatal care only is compensated at reduced rates, yet requires the provider to pay the same malpractice insurance premiums. Referral sources may be hard to find. Hospital closure or discontinuation of labor and delivery services is a devastating blow to a family physician practicing obstetrics or

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an obstetrician-gynecologist, usually resulting in termination of maternity care.19 Rural hospital closure is not uncommon, especially during periods of unfavorable economic performance due to decreasing reimbursement, lack of providers, loss of malpractice coverage, poor management, poor financial performance, poor service, or lack of funded patients. When hospitals cease providing obstetrical services, they almost never reinstitute those services. Hospitals may not be able to commit to obstetrical services or invest in advertisement, equipment, or adequate nursing staff. A lack of adequate anesthesia services makes obstetrical care impossible. If a hospital advertises obstetrical care, it must provide those services 24 hours a day. Inadequate blood bank services also limit the capability of physicians to practice obstetrics. Loss of labor and delivery services at a rural hospital can have financial implications for the hospital. Women control health care. If a woman’s obstetrical care is good, she will return for subsequent pregnancies, her gynecologic care, and care for her children, spouse, and extended family. If care was not good, she may not return; neither will her family. Government Regulations: Government regulations such as electronic medical records (EMRs), hospital computerized physician order entry (CPOE), meaningful use, Medicare Administrative Contractor (MAC) audits, and Recovery Audit Contractor (RAC) audits34,35 are a new challenge for physicians of all specialties. EMRs have been a challenge for the majority of physicians due to cost, decreased productivity, and a slow learning curve. Many have seen the EMR as a reason to quit practicing altogether. Technical Issues: High cesarean section rates may pose difficulties for physicians at peer review, quality improvement, and with insurance companies. Sometimes older physicians do not agree with current recommendations for management of breech presentation or vaginal birth after cesarean section. Physicians may not keep up with new advances or their hospital may not be able to afford newer technologies. With time, technical skills may wane and physicians may not appreciate their own limitations. Financial: A major reason that family physicians stop delivering babies is the cost of malpractice insurance.9,31,32 Obstetrics is a high-risk specialty and, even with an exemplary track record, premiums increase each year and usually plateau at year five. Plaintiff verdicts, settlements, multiple suits, and physician impairment can increase premiums. At some point physicians have to decide from a business standpoint if it is feasible to continue delivering babies.19 While it would be helpful to provide prenatal care in rural areas, many insurance carriers charge the same rates for partial prenatal care as they do for complete obstetrical care, making such partial care impossible financially. Payer mix, lack of commercial insurance patients, lack of public assistance patients, and decreased reimbursements contribute to decreasing obstetrical care. Better health care coverage for those in rural areas has allowed that subset of patients to travel to urban areas for care. Inadequate financial planning,

poor practice management, and decreased income contribute to physicians discontinuing obstetrics as well as practice. Political: Despite the lack of obstetrical providers nationwide, almost fifty percent of obstetricians do not support the idea of family physicians practicing obstetrics.1,15,19 Older OB/GYNs tend to support family physicians, but as they retire, who will take their place?15 Hospitals may decide not to allow family physicians obstetrical privileges, cesarean section privileges, or the ability to care for high-risk patients. The number of family physicians delivering babies continues to drop. So, one has to ask, if we do not have enough obstetrical providers, why would obstetrician-gynecologists oppose family physicians providing obstetrical care? Is it adequacy of training? Is it bad outcomes? Most likely, it is financial. Many obstetrician-gynecologists think it is probably acceptable for family physicians to deliver babies in rural areas but not metropolitan areas. Educational: Non-fellowship training and apprentice training may not be sufficient to practice obstetrics today. Insufficient patient numbers for training and inadequate breadth of training may not provide a physician with adequate education. Lack of continuing medical education contributes to physician inadequacy in practicing medicine, especially obstetrics. Sometimes physicians just do not “keep up.” In some cases, physicians did not learn basic components of practicing obstetrics, such as ultrasonography, instrumental deliveries, or surgical techniques. Patient numbers may be inadequate to afford malpractice insurance or to keep one’s skills current. Attrition: Many family physicians who offer obstetrical care practice for a long time and often in one locale. Due to age and declining health, there comes a time to either stop obstetrics or stop practicing altogether. The challenge is that the number of graduating residents is not keeping up with the number of retiring physicians. Occasionally, physicians move to another location or may be recruited to another location since family physicians are in great demand in this country. Family physicians are trained in many areas and often make a living in multiple areas even if they no longer deliver babies. Lack of Physician Support: Family physicians may find it difficult to find new physicians willing to practice obstetrics the way that they do. No help and no hope of help can be discouraging. Rural physicians practicing solo eventually wear out physically and mentally and feel that they can no longer practice obstetrics or even practice at all. Physicians need time off from on call, vacation time, and continuing medical education, not to mention hospitalization and surgery for themselves or immediate family members. Any time off requires call coverage, which can be difficult to obtain or unaffordable. Lack of call group support for obstetrics and loss of a partner can be major issues.19 Lack of OB/GYN back-up and lack of an OB/ GYN referral source at a larger center may discourage family physicians from practicing obstetrics, especially in a rural area. Lack of support and availability by other specialties, such as anesthesia, general surgery, internal medicine, and pediatrics, can have limiting effects on a family physician’s practice. While

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graduate obstetrician-gynecologists occasionally settle in rural areas, they seldom stay there very long.5 Most OB/GYN graduates migrate to urban areas after residency.1 Only half of obstetrician-gynecologists believe that family physicians should deliver babies.15 The half that do are the older physicians that are retiring.5 Family physicians preparing for careers in rural, underserved areas that sorely need maternity care need to be able to perform cesarean sections.36 Some rural areas may not be able to support an OB/GYN but could support two or three family physicians that provide obstetrical care.25

Recommendations and Considerations to Continue Practicing Obstetrics The importance of training family physicians to provide obstetrical care has never been more important. Graduating family medicine residents often lack the experience to practice in rural areas.18-23 There are a number of recommendations and considerations that may allow family physicians to continue practicing obstetrics. Family Physicians Are Trained to Provide Obstetrical Care: Obstetrics is part of the family medicine residency curriculum in which residents are trained to provide obstetrical care. Family medicine residency programs have always taught obstetrics with the assumption that some graduates would include maternity care as part of their practice. There are standard, twomonth rotations in OB/GYN, six-month obstetrical tracks for those interested in providing maternity care, and junior attendings that could perform cesarean sections with the presence of an attending physician.16,25 Enhanced obstetrical tracks have been successful in providing additional training in obstetrics. Family medicine obstetrics fellowships are designed to augment the obstetrical training already acquired during residency.17-21 Some components of maternity care may be part of many different residencies. Transitional residents may obtain two months of OB/GYN beyond medical school and are capable of performing vaginal deliveries. Pediatrics residents are trained to resuscitate and care for newborns. Some general surgery residents learn to perform cesarean sections. Emergency medicine residents learn how to perform spontaneous vaginal deliveries and resuscitate and immediately care for newborns. Most anesthesiology residents can intubate and resuscitate newborns. Certified OB/GYN nurse practitioners can manage prenatal care. Certified nurse-midwives can provide prenatal care and perform vaginal deliveries but cannot perform cesarean sections. Even plastic surgery fellows can perform a seven-layer closure of an episiotomy. Only obstetrician-gynecologists and family physicians are trained to perform the full spectrum of obstetrical care including prenatal care, labor, vaginal delivery, cesarean section, resuscitation of the newborn, and postpartum care. The scope of practice of a family physician is much broader than an OB/ GYN and includes newborn care.24,25 OB/GYNs only go in the nursery to visit newborns they have delivered or to perform a circumcision, and even that is decreasing.

Legal: Family physicians are almost never sued. They are at the opposite end of the spectrum of malpractice risk from OB/ GYNs.24 Obstetrician-gynecologists are in the highest malpractice risk category in most states. Family physicians often take care of the entire family in a long-term relationship. There is a stigma associated with suing a family doctor in a rural area; the fear is the doctor will leave and no other physician will be attracted to the area.24 Patients: All physicians have patients that are demanding and difficult to deal with. Sometimes it may even be necessary to terminate the physician-patient relationship. Early in the relationship, it is worthwhile to discuss patient expectations and realities, such as after-hours access, on call arrangements, and who may deliver their baby. Sorting this out at the first prenatal visit usually works out better than at the 39th week of gestation. Patients transfer to other providers for many reasons. If these transfers are excessive, it may be necessary for physicians to examine their mode of practice, office staff, telephone access, and even the hospital. A physician needs sufficient numbers to afford to practice obstetrics. Personal Health: Maintaining adequate health and reasonable work hours is essential to practicing medicine in general and, certainly, obstetrics. Reasonable work hours, call coverage, and time off allow physicians to maintain work-life balance. A community may be able to support two or three family physicians while it could support only a single OB/GYN with no call coverage.24 Two or three family physicians in a rural area can have a much better quality of life than a single provider, such as an OB/GYN. Physical exhaustion can lead to physician impairment. Collaborative arrangements for call relief, call coverage, vacation, illness, and continuing education can be lifesaving. Psychological: The fear of lawsuits is real; however, the odds of a family physician being sued are much lower than an OB/ GYN. Peer review and quality improvement are an integral part of medicine today and are here to stay. Collaboration with these groups will improve patient care and one’s practice in the long run. Mentoring new colleagues and, at the same time, learning new ideas and new techniques from them can be good for all concerned, including patients. There will always be a few patients that want to see the “new kid on the block.” Physicians should be open to lifelong learning and be willing to accept the fact that they may be at a disadvantage if there is something new that patients want or becomes the standard of care and they have not learned it. Feeling unappreciated or inadequate may be the result of exhaustion. While obstetrics can be stressful, it also has one of the highest levels of personal satisfaction in medicine due to bringing a new life into the world. Most family physicians that have embarked on training and practicing obstetrics have great personal satisfaction from delivering babies. Personal Life: Stressors at home are usually the result of not being at home. Arrangements for time off, continuing education, vacation, and professional counseling may help the situation at home. Collaborative agreements can also be helpful to a single provider. Such agreements can provide an avenue for

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Table 2: Recommendations & Considerations to Keep Family Physicians Delivering Babies

LEGAL

• Family physicians are almost never sued26 • Family physician malpractice rates cost less than rates for OB/GYNs26 • It makes good legal and financial sense to practice obstetrics

PATIENTS

• Clear practice policy for call, afterhours calls, prescriptions, etc. • Clear understanding of practice and call at initial visit • May need to evaluate self, office staff, etc.

PERSONAL HEALTH

PSYCHOLOGICAL

PERSONAL LIFE

TECHNICAL

• Know your own abilities and limitations • Experience has great worth with patients • Be involved with life-long learning • Continuing education is essential to staying current • Learn newer techniques or refer to those that do newer techniques • Make patient care the highest priority

FINANCIAL

• Malpractice rates for family medicine are a fraction of that for OB/GYN • Family physicians are rarely sued • Many states have reduced state income taxes for rural physicians • Many states have higher reimbursement for rural obstetric care • Medical scholarship programs forgive indebtedness for rural service

POLITICAL

• Adequate training, possibly obstetrics fellowship training, is important • Clear understanding of level of care to be provided is important • Collaborative agreements

• Maintaining adequate work hours, call, off hours • Collaborative arrangements for call, backup, transfer and referral24 • Maintaining personal health • Practice partners • Numerical odds of a suit are very low • Personal relationship with patient is everything • Involve self with peer review and quality assurance programs • Continue life-long learning • Mentor new physicians and learn from them • Obstetrics has one of the highest personal values of care • On call, vacation, and illness support is essential • Collaborative arrangements for call, backup, transfer, and referral • Professional counseling may be useful • Obstetrics has one of the highest personal values of care

• Personal relationship with patient is essential BAD • Family physicians are almost never sued OUTCOMES • Obstetrics has one of the highest personal values of care

HOSPITALS

• Meaningful use money to help defray EMR costs • Hospital programs to teach CPOE GOVERNMENT • MAC and RAC audits are in the REGULATIONS hospital arena but coming to physicians • Accurate billing, records, and coding are essential

• Obtain full obstetrics privileges including cesarean section • Resolution of privileges before contracts, moving, etc. • Contract needs to include support services like anesthesia • Clear lines of communication between hospital and physicians

• Adequate training and maybe obstetrics fellowship training • Board certification, recertification and maintenance certification EDUCATIONAL • Requirement for annual continuing education37 • Online educational programs

ATTRITION

• Encouragement of medical students to pursue family medicine • Encouragement of family medicine residents to practice obstetrics • Support of programs attracting physicians to rural areas

LACK OF PHYSICIAN SUPPORT

• Community can support 2 or 3 family physicians instead of 1 OB/ GYN24 • Collaborative agreements • Physician re-entry programs37-39

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

referrals and transfers for patients; coverage for time off, vacation, CME, and illness; or even relief from a large volume of patients. As stated above, obstetrics does have one of the highest levels of personal satisfaction. Bad Outcomes: Bad outcomes do occur even with the best of care in all specialties of medicine. Obstetrics is often highlighted because of a young mother or newborn baby in contrast to an elderly sick patient whose demise is expected. From a practical standpoint, family physicians are rarely sued because of their long-term relationships with patients and families. Hospitals: Hospital privileges, including cesarean section capability, must be resolved early in any agreement. Family physicians often have difficulty obtaining cesarean section privileges, and the ability to perform a cesarean section is essential to practicing modern day obstetrics. This should be resolved before a physician signs a contract and moves to a certain area. Commensurate with privileges is adequate hospital support with anesthesia services, nursing, blood bank, laboratory, etc. Call coverage and physician backup is also important. Even when all these issues have been resolved, hospitals still close obstetrical services, usually for financial reasons, or even close entirely. Everyone involved is affected when hospitals close: patients, physicians, employees, and the entire community. Clear lines of communication between hospital administration and physicians are essential. Government Regulations: Government programs, most of which are mandatory, are here to stay. Electronic medical records are not just a good idea, they are mandatory. There is meaningful use money available to help defray some of the cost of EMRs. Most hospitals have educational programs to teach CPOE to physicians, since hospitals stand to benefit the most from them. MAC and RAC audits are also here to stay. While hospitals are dealing with both now, these audits are coming to physicians’ offices in the near future. Accurate billing, records, and coding are essential to defend a physician’s work. Technical: Most technical issues fall into two areas: keeping up one’s skills and having the ability to learn new ones. Physicians should know their own abilities and limitations and make patient care the highest priority. Continuing education and sufficient patient numbers are important in keeping current skills sharp. To stay current, physicians must be willing to learn new techniques and adjust to the current standard of care. Experience has great worth with patients. Physicians should appreciate that small hospitals may not be able to provide all technological advances. If a physician cannot learn a newer technique, the physician should develop a referral plan to those who can offer the service. Financial: In many states, the cost of malpractice insurance for family physicians practicing obstetrics is significantly less of what OB/GYN physicians pay. Whereas a small community may not financially support an OB/GYN, it can support two or three family physicians that also practice obstetrics. It makes good business sense for family physicians to practice obstetrics. Malpractice costs less. Reimbursement is greater. Over-

head is significantly less. The addition of obstetrics to a family medicine practice usually includes only the costs of malpractice insurance and perhaps an ultrasound machine.24 In Alabama, there is a credit available that may reduce state income taxes for a physician practicing in a rural area. Some public assistance programs also reimburse physicians in rural areas at higher rates for prenatal care and delivery. Many medical scholarship programs forgive indebtedness associated with a doctor who practices in a rural, underserved area. Political: Many OB/GYNs feel that it is acceptable for family physicians to deliver babies in rural areas but not in urban areas. Most of the threat felt by OB/GYNs may be financial. Hospitals may not be able to support labor and delivery units or the cost of high-risk obstetrics. Clear understanding and agreement between physicians, nurses, and administration on which patients the hospital can care for and which patients need to be transferred is essential. Collaborative agreements for referral and transfer can help. Adequate training and probably obstetrical fellowship training is important for obtaining privileges. Educational: Adequate family medicine training and probably obstetrical fellowship training will be important in the future. Board certification, recertification, and maintenance of certification are also important in most hospitals. Many recertification programs can be completed online. Most states require annual continuing medical education to maintain privileges. Many states, including Alabama, have annual educational programs covering new and cutting edge information in obstetrics.37 There are many online educational programs available as well. Attrition: Growing older, giving up practice including obstetrics, and retiring is inevitable. Medical students who are interested in family medicine and possibly obstetrics need to be encouraged. It is essential to continue to recruit both family physicians and obstetrician-gynecologists to practice obstetrics. OB/GYNs and family physicians will have to work together to address the shortage of obstetrical providers. Lack of Physician Support: A community may be able to support two or three family physicians while it could only support a single OB/GYN with no call coverage.24 While OB/GYN physicians do occasionally go to rural, underserved areas, they rarely stay there very long.19 A family physician in a rural area provides many services including newborn care and care for the rest of the family.24 Two or three family physicians in a rural area can have a much better quality of life than a single provider. Family physicians often practice in rural areas.26 When call coverage is not available, collaborative arrangements can be made for transfers or practice coverage with general surgeons, experienced labor and delivery nurses, anesthesiologists, Certified Registered Nurse Anesthetists (CRNAs), gynecologists that have practiced obstetrics, paramedics, scrub technicians, and other medical specialists. Family medicine obstetrics blends these with the additional dimension of family support such as grandmothers. Physician re-entry into the workforce is a major emphasis of the American Congress of Obstetricians and Gynecologists.37-39

The Declining Number of Family Physicians Practicing Obstetrics: . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

There is limited data on physician re-entry into clinical practice, but OB/GYN physicians occasionally leave the practice of medicine for retraining, continuation, military service, and personal reasons which may also be applicable to family physicians.39 Availability of part-time work allows time for family needs. Since most OB/GYN residents today are women, the availability of part time work provides a new opportunity for those who wish to re-enter practice.39 With the declining number of physicians that provide obstetrical care in this country, any provider who wishes to re-enter practice and is willing to participate in the re-entry process is needed. The same would be applicable for family physicians that practice obstetrics. OB/GYNs and family physicians practicing obstetrics and working together is the only answer to addressing the shortage of obstetrics providers in this country.

Discussion There are many reasons that family physicians stop practicing obstetrics. Obstetrician-gynecologists occasionally stop delivering babies and attempt to make a living only practicing gynecology, usually unsuccessfully. Unless an OB/GYN has a special niche, practicing gynecology only is a difficult task. This is not the case with family medicine. Most family physicians can practice in many areas successfully without practicing obstetrics. Family physicians are often the only obstetrics providers in rural, underserved areas. Table 2 provides recommendations for support of family medicine with obstetrics. Graduating OB/GYN residents typically settle in urban areas. If they go to rural areas, they seldom stay there very long. There is not a shortage of OB/GYNs but only a maldistribution of them. The only group of obstetrical care providers that is growing is family physicians fellowship trained in obstetrics.17-21,36 OB/GYNs and family physicians practicing obstetrics should work together to address the shortage of obstetrical providers in this country. Family medicine physicians trained in obstetrical and newborn care is the answer to reducing maternal and perinatal morbidity and mortality in rural, underserved areas. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Daniel M. Avery, Jr, MD, is professor and chair of OB/GYN and professor and division chief of pathology at the University of Alabama School of Medicine in Tuscaloosa, AL. John T. McDonald, Jr, MD, CPA is an assistant professor of OB/GYN and clerkship director for OB/GYN at the University of Alabama School of Medicine in Tuscaloosa, AL.

References 1.

Rayburn WF, Klagholz JC, Murray-Krezan C, Dowell LE, Strunk AL. Distribution of American Congress of Obstetrician and Gynecologists fellows and junior fellows in practice in the United States. Obstet Gynecol.

2012;119(5):1017-22. 2.

Allen W. Obstetric care in a rural family practice. J Am Bd Fam Pract. 1989;2(1):30-3.

Reid AJ, Carroll JC, Ruderman J, Murray MA. Differences in intrapartum obstetric care provided to women at low risk by family physicians and obstetricians. CMAJ. 1989;140(6):625-33.

Rosenthal TC, Holden DM, Woodward W. Primary care obstetrics in rural western New York. A multi-center case review. N Y State J Med. 1990;90(11):537-40.

ME, Sills D, Connor PD. Perinatal outcomes: a comparison between family physicians and obstetricians. J Am Board Fam Pract. 1995;8(6):440-7.

Barclay AM, Knapp DP, Kallali KJ. The provision of delivery services by graduates of four Kansas family practice residencies. Kans Med. 1996;97(1):19-23.

Bowman RC. Continuing family medicine’s unique contribution to rural care. Am Fam Physician. 1996;54(2):471-4, 479, 483.

Loafman M, Nanda S. Who Will Deliver Our Babies? Crisis in the Workforce. Am J Clin Med. 2009;6(2):11-6.

Avery DM, Avery KT. Why do physicians stop practicing obstetrics? Medicolegal OB/GYN Newsletter. 2006.

10. Cohen D, Coco A. Declining trends in the provision of prenatal care visits by family physicians. Ann Fam Med. 2009;7(2):128-33. 11. Tong ST, Makaroff LA, Xierali IM, et al. Proportion of family physicians providing maternity care continues to decline. J Am Board Fam Med. 2012;25(3):270-1. 12. Blanchette H. The impending crisis in the decline of family physicians providing maternity care. J Am Board Fam Med. 2012;25(3):272-3. 13. Loy CS, Warton RB, Dunbar JA. Workforce trends in specialist and GP obstetric practice in Victoria.Med J of Aust. 2007;186(1):26-30. 14. Rodney WM. Obstetrics in family practice. J Am Board Fam Pract. 2002;15(3):255-6. 15. Topping DB, Hueston WJ, MacGilvray P. Family physicians delivering babies: what do obstetricians think? Fam Med. 2003;25(10):737-41. 16. Coonrod A, Kelly BF, Ellert W, Loeliger SF, Rodney WM, Deutchman M. Tiered maternity care training in family medicine. Fam Med. 2011; 43(9):631-7. 17. Peterson LE, Blackburn B, Phillips RL, Puffer JC. Structure and characteristics of family medicine maternity care fellowships. Fam Med. 2014;46(5):354-9. 18. Norris TE, Acosta DA. A fellowship in rural medicine: program development and outcomes. Fam Med. 1997;29(6):414-20. 19. Rodney WM, Martinez C, Collins M, Laurence G, Pean C, Stallings J. OB fellowship outcomes 1992-2010: where do they go, who stops delivering, and why? Fam Med. 2010;42(10):712-6. 20. Eidson-Ton WS, Nuovo J, Solis B, Ewing K, Diaz H, Smith LH. An enhanced obstetrics track for a family practice residency program: results from the first 6 years. J Am Bd Fam Pract. 2005;18(3):223-8. 21. Chang PC, Leeman L, Wilkinson J. Family medicine obstetrics fellowship graduates: training and post-fellowship experience. Fam Med. 2008;40(5):326-30. 22. Bowman RC: Rural practice and obstetrics fellowships for family physicians [Letter to the Editor reply]. Am Fam Physician. 1997;55(8):2603-4. 23. Acosta DA. Impact of rural training on physician workforce: the role of postresidency education. J Rural Health. 2000;16(3):254-61.

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

24. Avery DM. Why family physicians should practice obstetrics. Am J Clin Med. 2008;5(1):32-4.

32. Norris TE. Family practice OB: to be or not to be? Liability is the question. J Am Board Fam Pract. 2003;16(6):565-6.

25. Rodney WM, Crown LA, Hahn R, Martin J. Enhancing the family medicine curriculum in deliveries and emergency medicine as a way of developing a rural teaching site. Fam Med. 1998;30(10):712-9.

33. Nicholson JM. Risk adjustment in maternity care: the use of indirect standardization. Int J Women’s Health. 2010;2:255-62.

26. Hueston WJ. Specialty differences in primary cesarean section rates in a rural hospital. Fam Pract Res J. 1992;12(3):245-53. 27. Norris TE, Pirani MJ, Reese JW, Rosenblatt RA. Are rural family physicians comfortable performing cesarean sections? J Fam Pract. 1996;43(5):455-60. 28. Hueston WJ, Lewis-stevenson S. Provider distribution and variations in statewide cesarean section rates. J Community Health. 2001;26(1):1-10.

34. Compliance 360 Claims Audit Manager for CMS Medicare RAC Audits. Managing RAC, MAC and other Medical Claims Audits. Compliance 360 Website. http://www.compliance360.com/RAC/ 35. CMS Announces New Recovery Audit Contractor Program to identify improper Medicare payments. Centers for Medicare & Medicaid Services Web site. http://www.cms.gov/Newsroom/MediaReleaseDatabase/FactSheets/2008-Fact-Sheets-Items/2008-10-06.html. 36. Avery DM. A new certification for FPs. J Fam Pract. 2011; 60(3):E1-3.

29. Hueston WJ. Site-to-site variation in the factors affecting cesarean section rates. Arch Fam Med. 1995;4:346-51.

37. Cass I, Gordon LA, Kilpatrick S. Lessons learned in physician reentry. Obstet Gynecol. 2012;120(2 Pt 1):360-4.

30. Fisher TK. Maternity Care Work Force Analysis. CPM Symposium. March 18, 2012. Adapted from Rayburn WF. The Obstetrician-Gynecologist Workforce in the United States: Facts and Implications. 2011.

38. Martin JN, Scarrow PK. Reentering obstetrics and gynecology. Obstet Gynecol. 2012;120(2 Pt 1):217-8.

31. Larimore WL, Sapolsky BS. Maternity care in family medicine: economics and malpractice. J Fam Prac. 1995;40(2):153-60.

39. Manriquez M, Cookingham LM, Coonrod DV. Reentry into clinical practice in obstetrics and gynecology. Obstet Gynecol. 2012;120(2 Pt 1):364-9.

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The Declining Number of Family Physicians Practicing Obstetrics: . . .

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

MEDICAL ETHICS WITHOUT THE RHETORIC

Cases presented here involve real physicians and patients. Unlike the cases in medical ethics textbooks, these cases seldom involve human cloning, bizarre treatments, or stem cell research. We review cases more common to the practice of medicine. Most cases are circumstantially unique and require the viewpoints of the practitioners and patients involved. For this reason, I solicit your input on the cases discussed here at councile@aol.com. Reader perspectives along with my own viewpoint are published in the issue following each case presentation. We are also interested in cases submitted by readers. The following case addresses an ethical issue that arises when a physician has another physician as a patient. Mark Pastin, PhD Mark Pastin, PhD, is president and CEO of the Council of Ethical Organizations, Alexandria, VA. The Council, a non-profit, non-partisan organization, is dedicated to promoting ethical and legal conduct in business, government, and the professions.

CASE FIFTEEN

- SHAKY GROUNDS

Your patient is a highly regarded surgeon who is a member of the same medical staff to which you belong. She complains that she is occasionally bothered by shaky hands and blacked out for just a second once during surgery. You think she smells of alcohol and that alcohol abuse is probably the problem. When you raise this issue, she flatly denies having alcohol problems and insists that it is some sort of neurological problem. You are skeptical but agree to run some tests on the slight chance she is right. When you see her a week later, you report that all the tests have come back negative. You can see no other cause of her problem except alcohol or drugs. You discretely ask about her at the hospital, but no one has noticed anything but excellent performance from her. Your concern is that a patient could be seriously injured if she blacks out or has an attack of the shakes during surgery. You wonder if you have an obligation to report your concern to the medical staff or hospital administration. You are also concerned about maintaining patient privacy. Additionally, if you report the surgeon, others having substance abuse issues may be unlikely to seek medical attention.

This is an actual case. Of course, there are any number of complicating circumstances and additional details; but please address the case on the basis of the information provided. There will be an analysis of this case and a new case in the next issue.

Your input is requested. Email your responses to: councile@aol.com. © Copyright Council of Ethical Organizations 2014

Medical Ethics Without the Rhetoric

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

MEDICAL ETHICS WITHOUT THE RHETORIC

CASE FOURTEEN ANALYSIS -FREQUENT FLYER

The case from the last issue involves a patient who comes to you because he is having anxiety attacks. This patient is notorious for seeing multiple physicians with the same complaint, even if the complaint is fanciful. In this case, the patient reports that he saw a psychiatrist the day before the visit with you. The patient complains that the psychiatrist gave him a prescription, but he is still having anxiety attacks. You look at the prescribed medicine and realize that it is a multi-vitamin. The question is whether to tell the patient. Most readers took the position that they would not tell the patient that he was taking what is probably a placebo because there is a slight chance that a vitamin deficiency is related to the reported anxiety attacks. Most would suggest that the patient go back to psychiatrist and report that he is not improving. However, given the patient’s history, there is every chance that he will just seek the advice of yet another medical practitioner. Many readers would suggest that the patient continue following the psychiatrist’s instructions, but prescribe an anti-anxiety medication - just in case the patient’s complaint is real. Most readers would also advise the patient to select one medical practitioner and follow the advice they receive from that practitioner - thereby inviting the patient not to return under similar circumstances. My own view of this case is that the patient should be informed that he is taking a multi-vitamin in the interests of providing honest information to someone who is, at least at the moment, your patient. I would follow readers in recommending that he go back to the psychiatrist with his problem and focus on following one doctor’s advice.

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

A Case Report on the Recurrence of Lemierre’s Syndrome: Awareness to Diagnosis and Treatment Caleb A. Awoniyi, MD, PhD Douglas Hanson, MD

Abstract In 1936, Andrew Lemierre described a syndrome that now bears his name. He presented 20 cases of postanginal septicemia in healthy young adults that involved an acute oropharyngeal infection complicated by septic thrombophlebitis and metastatic infection.1,2 In recent years, more cases of Lemierre’s syndrome have re-emerged, thus bringing back into the spotlight the importance of prompt diagnosis and treatment.3 Because of its previous rarity, clinicians may overlook and miss the diagnosis at the initial presentation. Mortality has been estimated at 15% to 18%, and the disease itself has been associated with significant morbidity.4,5 It is crucial that clinicians have a high index of suspicion and heightened awareness of signs and symptoms associated with this disease. Early recognition and treatment invariably leads to optimal outcome.

Introduction Lemierre’s syndrome was common in the era before the advent of antibiotics. In the 1960s and 1970s, Lemierre’s syndrome became a rare phenomenon with the proliferation of penicillin in the treatment of pharyngitis. Today, the incidence of Lemierre’s syndrome is estimated to be 1 per million annually.6 The judicious use of antibiotics has led to an increase in the incidence of Lemierre’s syndrome. The main etiological agent in Lemierre’s syndrome is Bacillus fundiliformis, now known as Fusobacterium necrophorum, though other species such as Streptococcus, Bacteroides, Lactobacillus have also been implicated.2,7 F. necrophorum is a part of the normal flora of the oral cavity, GI tract and female genital tract. It is a non-motile,

gram-negative, obligate anaerobic rod that is characteristically pleomorphic with filaments.6 The oropharynx is the primary site of infection in Lemierre’s syndrome, specifically the lateral pharyngeal space.7-9 However, nasopharyngeal infection, mastoiditis, otitis media, sinusitis, and odontogenic infection as initiating sites have also been reported. 7-9 The first stage of the primary infection is usually bacterial or viral pharyngitis, such as streptococcal pharyngitis or Epstein-Barr virus mononucleosis.6,7,9,10 It is postulated that the antecedent infection creates a favorable environment for Fusobacterium species to proliferate within the oropharyngeal space via immunosuppression and disruption of mucosal barriers.11,12 The pathogenesis of septic thrombophlebitis is best understood in the context of Virchow’s triad. Given the proximity of the lateral pharyngeal space to the internal jugular vein, the bacteria invade the internal jugular vein either by direct extension or lymphatic and/or hematological extension. The resultant damage to the endothelial wall of the internal jugular vein produces a change in the vessel wall that alters the pattern of blood flow, leading to hypercoagulability and development of thrombosis. Metastatic infections to other organs can then ensue.11,12

Case Presentation: A 19-year-old male with relatively benign medical history presented to the emergency department with complaints of shortness of breath and right-sided abdominal pain. He had been healthy until one week prior when he initially presented to a walk-in clinic with fatigue, sore throat, and vague myalgias. At that time, he had a positive Monospot test and was diagnosed

A Case Report on the Recurrence of Lemierre’s Syndrome . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

with mononucleosis. He was released with supportive measures. He was stable for a few days but then started feeling increasingly fatigued and lethargic. He had decreased oral intake and some episodes of non-bloody emesis. The day before admission, he noticed dark colored urine as well as a change in his skin color to a yellowish hue. On the day of admission to the emergency department, he became more short of breath with increasing primarily right-sided abdominal pain. His complaints included vague abdominal pain, shortness of breath, myalgias, fever, headache, and weakness. He denied chest pain, palpitations, dysuria, melena, hematochezia or any neurologic deficits.

There was no jugular venous distention. He was tachycardic but otherwise normal sinus rhythm without murmur. He had bilateral wheezes with diminished breath sounds and increased coarseness in the right lung field. Abdomen exhibited diffuse tenderness with mild rebound tenderness but was without distention or guarding. Hypoactive bowel sounds were also noted. There was no evidence of ascites or organomegaly. He had a positive heal tap and iliopsoas test. Peripheral pulses were palpable but with mildly delayed capillary refill. Cranial nerves II-XII were grossly intact bilaterally. There were no focal areas of weakness, numbness, or paresthesia.

Figure 1A: Chest x-ray on admission indicating ARDS

Figure 1C: CT scan showing iliopsoas abscess – arrow

Figure 1B: CT scan of chest with contrast showing several cavitary emboli and pleural effusion

Since the patient appeared to be volume depleted in the emergency room, he was given intravenous fluids. After receiving 1200 mL of normal saline, he became increasingly short of breath and oxygen saturation by pulse oximetry subsequently dropped into the mid-80s. He was immediately given oxygen via a 100% non-rebreather mask to maintain saturation greater than 90%. A chest x-ray revealed diffuse infiltrates resembling acute respiratory distress syndrome or cardiogenic pulmonary edema (Figure 1A). Abdominal films showed mildly dilated loops of bowel consistent with possible ileus; however, no evidence of frank obstruction was noted. Chest and abdominal computed tomography (CT ) scan with and without oral contrast demonstrated small, nonspecific cavitary pulmonary nodules resembling septic emboli and right pleural effusion (Figure 1B). CT scan also revealed a 3.2 cm mass and some free air within the iliopsoas consistent with an abscess cavity (Figure 1C and D), splenomegaly, and moderate degree of ascites throughout the abdomen and pelvis. The patient was admitted to the intensive care unit and started empirically on ceftriaxone and piperacillin/tazobactam. He grew increasingly hypoxic and tachypneic, with respiratory failure progressing to intubation. By the third day of hospitalization, his condition had improved and he was extubated. His blood culture grew Fusobacterium necrophorum. Due to mild left neck pain, a CT scan of his head and neck was obtained, revealing occlusion of

On evaluation, he had a blood pressure of 124/95, heart rate of 116, and respiratory rate was 32. Pulse oximetry reading was 94% on room air. He was thin, jaundiced, and appeared to be in mild respiratory distress. He had mild scleral icterus bilaterally. Oropharynx was dry but otherwise non-erythematous.

A Case Report on the Recurrence of Lemierre’s Syndrome . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

left internal jugular vein (IJV) (Figure 1E). This constellation of symptoms – respiratory distress, pulmonary cavitary nodules with pleural effusion, iliopsoas abscess, occlusion of internal jugular, and bacteremia with Fusobacterium necrophorum are consistent with Lemierre’s syndrome. Given concerns for cerebrospinal fluid (CSF) disease (~3-4% in Lemierre’s syndrome) and lack of CSF penetration from piperacillin/tazobactam, antibiotic coverage was changed to metronidazole. The patient’s fever persisted for more than 96 hours, increasing concerns regarding the iliopsoas abscess. A decision was ultimately made not to drain the abscess based on the patient’s elevated international normalized ratio (INR) and the potential for hematoma and/or osteomyelitis. The patient’s fever resolved on day 6 of hospitalization. His condition improved and he was discharged on day 16 of hospitalization with a plan to continue antibiotics for an additional six weeks. The patient was scheduled to follow up in a week with a chest x-ray and pelvic CT scan to re-evaluate his iliopsoas abscess. Figure 1D: CT scan showing iliopsoas abscess with some free air –

arrow

Diagnosis and Clinical Features The main features of Lemierre’s syndrome include: (1) primary infection of oropharynx, (2) septicemia documented by positive blood culture, (3) septic thrombophlebitis of the IJV confirmed by radiological evidence, and (4) metastatic septic emboli. The septic emboli usually consist of pulmonary foci but also can involve the joints, liver, spleen, bones, kidneys, and meninges.8,11,13 The diagnosis is typically made on clinical grounds with high index of suspicion. However, more often than not, this is not thought of until the bacteria are isolated from culture. Patients are typically young healthy adults, although cases have been observed in all age groups.4,11,14 Patients characteristically present within one week after onset of tonsillitis or pharyngitis with persistent fever, intense rigors, ipsilateral neck pain, and swelling along the anterior border of the sternocleidomastoid muscle. In this case report, the initial diagnosis was mononucleosis, but the patient returned a few days later with fever and respiratory failure, likely due to septic pulmonary emboli. Other associated symptoms of Lemierre’s syndrome include myalgia, arthralgia, diffuse abdominal pain, and more commonly, pulmonary involvement such as productive cough, dyspnea, pleuritic chest pain, and/or hemoptysis. Laboratory findings include moderate leukocytosis with left shift, thrombocytopenia, mildly elevated liver enzymes, coagulopathy consistent with disseminated intravascular coagulation (DIC) clinical picture, and electrolyte abnormalities consistent with syndrome of inappropriate secretion of antidiuretic hormone (SIADH). In addition, the erythrocyte sedimentation rate is always elevated.4-8,13 Radiological studies are key in establishing the diagnosis of thrombosis to the internal jugular vein. Contrast-enhanced CT scan is the modality of choice, although others have used ultrasonography. The drawback in using ultrasound is that it provides poor imaging beneath the mandible and the clavicle and can miss fresh thrombus with low echogenicity.8,13 In the face of pulmonary involvement, chest x-ray may show associated pleural effusion and bilateral, nodular cavitation.8

Treatment Figure 1E: CT scan of neck showing occlusion of the internal

jugular vein – arrow

The mainstay of therapy is antibiotics. The isolation of F. necrophorum can take up to several weeks. Antibiotic treatment should not be delayed until the blood culture results are available. F. necrophorum is generally susceptible to penicillin, metronidazole, and clindamycin.8,12,14 However, treatment failures with penicillin have been reported due to B-lactamase-resistant strains of Fusobacterium. As such, B-lactamase-resistant antibiotics with anaerobic activity are recommended.7 Due to the potential for polymicrobial involvement, combinations of antibiotics are often used.13 Treatment duration is guided by clinical response. Typically, parental antibiotic treatment is required in the initial phase, usually from 1-2 weeks. This is followed by another 2-4 weeks of oral therapy.14 Surgical drainage of abscess may be required, particularly in the face of failing medical treatment. The role of anticoagulation in the treatment of septic thrombophlebitis of the IJV remains contro-

A Case Report on the Recurrence of Lemierre’s Syndrome . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

versial.5 The main concern with the use of anticoagulation is that it may facilitate embolic spread of the infection.6 Others have advocated the use of anticoagulation if the thrombosis propagates to the cavernous sinus or in cases of completely vaso-occlusive jugular vein thrombosis.15,16 Surgical ligation of thrombosed IJV or venous resection is advocated in rare cases when medical therapy has failed.18

Conclusion As was rightly stated by Hall, “Be not deceived by a comparatively innocent appearing pharynx, as the veins of the pharynx may be carrying the death sentence for your patient.” The prognosis of Lemierre’s syndrome is related to prompt initiation of therapy. A worsening of pharyngitis or mononucleosis with fever, chill, and/or respiratory failure a few days or weeks after presentation should raise a high index of suspicion for Lemierre’s syndrome. Constellations of symptoms including pharyngitis (bacterial or viral), neck pain, and systemic illness are classic for this disease and should be undoubtedly included in the differential diagnosis. Once suspected, initiation of antibiotics should not be delayed until culture result is available. Anticoagulation should be considered as well as surgical drainage for metastatic abscesses. 19

Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Caleb A. Awoniyi, M.D, PhD, is a staff physician for North Florida/South Georgia Veterans Health System in Gainesville, FL. Douglas Hanson, MD, is a staff physician in the Department of Medicine at Broadlawns Medical Center in Des Moines, IA.

References 1.

Lemierre A. On certain septicemias due to anaerobic organisms. Lancet. 1936;1:701-3.

Moore BA, Dekle C, Werkhaven J. Bilateral Lemierre’s syndrome: A case report and literature review. Ear Nose Throat J. 2002;81:234-52.

Kushawaha A, Popalsai M, El-Charabaty E, Mobaraka N. Lemierre’s syndrome, reemergence of a forgotten disease: a case report. Cases J. 2009;2:6397-00.

Karkos PD, Karkanevatos A, Panagea S, Dingle A, Davies JE. Lemierre’s syndrome: how a sore throat can end in disaster. Eur J Emerg Med. 2004;11:228-30.

Bliss SJ, Flanders SA, Saint S. A pain in the neck. N Engl J Med. 2004;350:1037-42.

Chirinos JA, Lichtstein DM, Garcia J, Tamariz LJ. The evolution of Lemierre syndrome. Medicine (Baltimore). 2002;81:458-65.

Hagelskjaer LH, Prag J. Human Necrobacillosis, with emphasis on Lemierre’s syndrome. Clin Infect Dis. 2000;31:524-32.

Golpe R, Marin B, Alonso M. Lemierre’s syndrome (necrobacillosis). Postgrad Med J. 1999;75:141-4.

Dool H, Soetekouw R, Van Zanten M, Grooters E. Lemierre’s syndrome: three cases and a review. Eur Arch Otorhinolaryngol. 2005;262:651-4.

10. Love WE, Zaccheo MV. Lemierre’s syndrome: More judicious antibiotic prescribing habits may lead to the clinical reappearance of this often forgotten disease. Am J Med. 2006;119:E7-E9. 11. Venglarcik J. Lemierre’s syndrome. Pediatr Infect Dis J. 2003;22:921-4. 12. Venkateswaran S, Sze FKH. A case of Lemierre’s syndrome presenting with multiple pulmonary abcesses associated with tension hydropneumothorax resulting in mediastinal shift. Ann Acad Med Singapore. 2005;34:450-3. 13. Riordan T, Wilson M. Lemierre’s syndrome: more than a historical curiosa. Postgrad Med J. 2004;80:328-34. 14. Ochoa R, Goldstein J, Rubin R. Clinicopathological conference: Lemierre’s syndrome. Acad Emerg Med. 2005;12:152-57. 15. Batty A, Wren MWD, Gal M. Fusobacterium necrophorum as the cause of recurrent sore throat: comparison of isolates from persistent sore throat syndrome and Lemierre’s disease. J Infect. 2005;51:299-306. 16. Goldenberg NA, Knapp-Clevenger R, Hays T, Manco-Johnson MJ. Lemierre’s and Lemierre’s-like syndromes in children: survival and thromboembolic outcomes. Pediatrics. 2005;116:e543-e8. 17. Repanos C, Chadha NK, Griffiths MV. Sigmoid sinus thrombosis secondary to Lemierre’s syndrome. Ear Nose Throat J. 2006;85:98-101. 18. Charles K, Flinn WR, Neschis DG. Lemierre’s syndrome: A potentially fatal complication that may require vascular surgical intervention. J Vasc Surg. 2005;42:1023-5. 19. Hall C. Sepsis following pharyngeal infections. Laryngol. 1939;48:905-25.

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A Case Report on the Recurrence of Lemierre’s Syndrome . . .

JUNE

2015

House of Delegates

26 thJULY st 1 2015

Annual Scientific Meeting

Changing Trends in Medicine HILTON CLEARWATER BEACH Âť CLEARWATER, FLORIDA

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

Extraosseous Ewing Sarcoma of the Larynx: A Report of a Rare Case Nicole F. De Simone, DO, MPH, Capt, USAF Anuj Parikh, DO Kurt Graupensperger, DO James P. Malone, MD

Abstract Objectives: Extraosseous Ewing sarcoma is a variant of Ewing sarcoma that can occur anywhere in the body. There are few documented cases reported in the head and neck region and only two reported cases arising from the larynx. Methods/Results: We report a case of a 45-year-old female who was found to have primary extraosseous Ewing sarcoma of the larynx. Initial workup of her new neck mass, appearing to arise from the thyroid gland, was concerning for lymphoma based on fine needle aspiration. However, further biopsy and immunohistochemistry confirmed a much different diagnosis. She received the current recommended therapy for this rare disease that has a high rate of recurrence. Conclusions: These tumors can be mistaken for other neoplasms due to their primitive morphology and rare occurrence. We discuss the workup to allow for accurate diagnosis, treatment, and prognosis to improve morbidity and mortality from this rare disease.

Introduction Ewing sarcoma (ES) is a rare, high-grade, malignant neoplasm composed of small round blue cells that originate from mesenchymal stem cells and commonly present in the skeletal system. ES is the second most common bone cancer, typically presenting in children, adolescents, and young adults.1-3 Rarely, it can arise in extraosseous sites, such as the lung, skin, pelvic cavity, retroperitoneum, and the head and neck region.4,5 Extraosseous Ewing sarcoma (EES) accounts for 1.1% of malignant soft tis-

sue tumors with 9% of them localized to the head and neck. Of these, 1-4% are diagnosed in otolaryngeal areas.4,6 The unusual site of involvement combined with the primitive morphology of EES can make diagnostic accuracy challenging, as it often mimics other neoplasms.4,7 To our knowledge, there have been only two previously reported cases of EES involving the larynx.3 We report a rare case of a 45-year-old female diagnosed with primary EES arising from the right larynx, initially thought to be lymphoma on outpatient evaluation of her neck mass. The diagnosis, pathogenesis, treatment, and literature review is discussed below.

Case Report A 45-year-old white female presented for routine follow-up for breast cancer to her oncologist. She developed a new anterior neck mass that gradually increased in size over a one-month period. The mass seemed to arise from her thyroid gland and was associated with hoarseness. The remainder of her clinical history was unremarkable. Her past medical history is significant for invasive ductal breast carcinoma which was diagnosed three years prior. She underwent a modified radical mastectomy and completed a course of adjuvant chemotherapy with doxorubicin and cyclophosphamide followed by paclitaxel. This was followed by adjuvant tamoxifen planned for five years. Physical examination of her neck revealed smooth, rubbery, non-tender masses bilaterally without palpable lymphadenopathy. Nasopharyngolaryngoscopy demonstrated complete unilateral right vocal cord paralysis with associated edema.

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A thyroid ultrasound showed a 2.9-cm complex, hypoechoic mass in the right upper pole of the thyroid, a separate 1.9-cm nodule anterior to this, and a 7 mm nodule in the left lower pole. All three nodules appeared to arise from thyroid tissue. Ultrasound-guided fine-needle aspiration of the right dominant nodule revealed atypical lymphoid elements suspicious for lymphoma. A subsequent positron emission tomography/computed tomography (PET/CT) was then obtained revealing two masses adjacent to, but distinct from, the thyroid gland, both of which were intensely fluorodeoxyglucose (FDG) avid. There were no sites of metastatic disease identified.

there was a new FDG avid spot on the sacrum (S1). Repeat nasopharyngolaryngoscopy demonstrated resolution of her laryngeal mass. Further MRI evaluation of the pelvis confirmed a 3.3 x 1.6-cm area of marrow replacement as well as cortical breakthrough of S1, concerning for metastatic disease. Figure 2: Cell block demonstrating tumor necrosis

Figure 1: Hematoxylin and eosin stain of cell block showing small round blue cell tumor with scant cytoplasm

Discussion

Additional tissue sampling was recommended to confirm the diagnosis of lymphoma; therefore, an excisional biopsy of the right thyroid mass was performed. Final pathology was consistent with EES. Neoplastic cells were positive for cluster of differentiation 99 (CD99), FL1-1 and weakly positive for synaptophysin. Fluorescence in situ hybridization (FISH) was positive for t(11;22). Sections demonstrated diffuse proliferation of small round blue cells with scant cytoplasm, inconspicuous nucleoli, and necrosis (Figure 1, Figure 2). Flow cytometry was negative for lymphoma and thyroid tissue. The patient was referred to a tertiary care facility for further treatment. Within one month of her biopsy, the neck lesions had grown significantly with worsening hoarseness. She then underwent pan endoscopy and biopsy of a lesion that clearly emanated from the right larynx. This biopsy was also consistent with EES. She immediately began treatment with four alternating cycles of modified vincristine, doxorubicin, and cyclophosphamide (VDC) with alternating ifosfamide and etoposide (IE) given over four days instead of the usual two to avoid cardiotoxicity given her prior doxorubicin exposure. She went on to receive two cycles of concurrent radiation as well as two additional cycles of IE. Near resolution of her hoarseness was achieved with induction of chemotherapy. Three months after her final radiation treatment, follow-up PET/CT showed significant volumetric and metabolic improvement in the perilaryngeal soft tissues when compared to prior PET/CT; however,

Tefft et al first described EES in 1969 as histologically similar to primary ES of the bone, usually presenting in the lower limbs or paravertebral area.3,4,6,8 EES is a rare, rapidly growing round cell malignant tumor that can develop in soft tissues at any location. Approximately 80% of ES cases will occur in patients less than 20 years of age, and 80% of these cases will arise from bone, primarily affecting the diaphysis or metaphysealdiaphyseal regions.7 In contrast, 50% or more of primary adult cases are extraosseous. For patients diagnosed with EES, approximately 15-46% will present with gross metastatic disease at the time of diagnosis. The most common site of metastasis for EES is the lung, followed by bone which has a particularly poor prognostic factor.9 The histological pattern of EES consists of solid sheets of small, round, uniform, primitive cells with round nuclei and scant cytoplasm that lack significant differentiation. These features make EES difficult to distinguish from other tumors such as primitive neuroepithelial tumor, rhabdomyosarcoma, neuroblastoma, small cell carcinoma, and malignant lymphoma.3,4,7 Immunohistochemical expression of CD99 is helpful in separating this entity from other small round cell tumors; however, it can also be expressed by other sarcomas.7 Molecular demonstration of the EWS-FL1 gene rearrangement via a FISH study is highly specific for EES and is present in 90-95% of these sarcomas, making this essential for accurate diagnosis. Immunohistochemistry shows positive prostate-specific antigen (PSA), CD99, vimentin, various expressions of neuron-specific enolase (NSE), synaptophysin (SYP), and cluster of differentiation 57 (CD57) and is negative for 100% soluble proteins (S-100) and nuclear factor (NF).2-7

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

Current treatment recommendations by the National Comprehensive Cancer Network (NCCN) for EES are the same as those for bone ES, with the main treatment being chemotherapy, surgery, and radiation.10 It has been shown that alternating VDC/ IE chemotherapy can improve the metastasis-free survival.4,6,10 Wide resection margins of 2-3 cm in conjunction with multiagent chemotherapy are necessary for good clinical outcomes.6 Radiotherapy has been regarded as the standard treatment for localized tumor therapy for decades. However, recent data advocates surgery when complete resection is possible.2 Induction chemotherapy is commonly used due to the high risk of recurrence and metastatic disease and should be given 12-24 weeks before surgery; additional post-operative chemotherapy and radiation is the treatment of choice when possible.1,4,6 Primary and metastatic ES are unique among sarcomas in that they can respond dramatically to initial therapy.1,9

Nicole F. De Simone, DO, MPH, Capt, USAF is a general surgeon at Joint Base Langley-Eustis Langley AFB, VA.

Gradoni P, Giordano D, Oretti G, et al. Clinical outcomes of rhabdomyosarcoma and Ewing’s sarcoma of the head and neck in children. Auris Nasus Larynx. 2011;38:480-6.

Despite good initial responses to treatment, EES usually follows an aggressive course with a high rate of both local recurrence and distant metastasis.4,9 Age and surgical treatment were found to be the most important prognostic indicators for response, followed by tumor location, tumor size, presence of metastasis, genetic mutation type, and treatment regimen.1,6,8 One study found the overall survival for 3 and 5 years to be 47% and 24%, respectively, when metastatic disease was present. The 3- and 5-year survival rates improved to 60% and 30%, respectively, when no metastasis was present.4 Another study showed that 30% of patients relapse, and of these patients, 2025% survived, making it critical that cure rates are improved.2

Ross KA, Smyth NA, Murawski CD, Kennedy JG. The Biology of Ewing Sarcoma. ISRN Oncol. 2013;2013:759725.

Yang YS, Hong KH. Extraskeletal Ewing’s sarcoma of the larynx. J Laryngol Otol. 2004;118(1):62-4.

Osborn HA, Wehrli B, Franklin JH, Fung K. Ewing’s sarcoma of the masseter muscle. J Laryngol Otol. 2011;125:978-81.

Maldi E, Monga G, Rossi D, Tosoni A, Mezzapelle R, Boldorini R. Extra-osseous Ewing sarcoma of the thyroid gland mimicking lymphoma recurrence: A case report. Pathol Res Pract. 2012;208(6):356-9.

Xie CF, Liu MZ, Xi M. Extraskeletal Ewing’s sarcoma: a report of 18 cases and literature review. Chin J Cancer 2010;29(4):420-4.

Manduch M, Dexter DF, Ellis PM, Reid K, Isotalo PA. Extraskeletal Ewing’s sarcoma/primitive neuroectodermal tumor of the posterior mediastinum with t(11;22)(q24;q12). Tumori. 2008;94(6):888-91.

Adapa P, Chung TW, Popck EJ, Hunter JV. Extraosseous Ewing sarcoma of the thyroid gland. Pediatr Radiol. 2009;39(12):1365-68.

Javery O, Krajewski K, O’Regan K, et al. A to Z of extraskeletal Ewing sarcoma family of tumors in adults: imaging features of primary disease, metastatic matterns, and treatment responses. AJR Am J Roentgenol. 2011;197:1015-22.

The imaging of EES is different from that of bone ES since they originate from different sites. Although imaging is non-specific, EES tends to be well circumscribed and of generally low attenuation on CT. It is commonly hypoechoic on ultrasound and vascularity is variable.4 Since this disease has a high rate of distant metastasis, the most common sites being lung and bone, it is suggested that chest radiograph or CT and bone scan be done before treatment to exclude distant metastasis and make a clinical stage with PET/CT holding promise for surveillance.

Anuj Parikh, DO, is a general surgery resident at Pinnacle Health System in Harrisburg, PA. Kurt Graupensperger, DO, is a general surgery resident at Pinnacle Health System in Harrisburg, PA. James P. Malone, MD, is an attending surgeon at Pinnacle Health System in Harrisburg, PA.

References

10. Ewing’s Sarcoma Family of Tumors Version 2.2013. National Comprehensive Cancer Network Web site. http://www.nccn.org. Accessed June 1, 2013.

Conclusion In conclusion, EES poses a diagnostic challenge for physicians. It is often mistaken for other neoplasms, which may delay diagnosis and worsen patient prognosis. Treatment includes neoadjuvant chemotherapy and wide surgical resection, followed by adjuvant chemotherapy and radiation. EES has a high rate of recurrence so surveillance imaging with PET/CT is recommended. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Extraosseous Ewing Sarcoma of the Larynx . . .

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

Undulant Fever in ED: A Case Report Andrzej Skarpetowski, MD

Abstract Brucellosis is the most common zoonosis worldwide. It is an uncommon entity in humans in the United States, with Texas reporting a third of cases. It is difficult to diagnose and hence can be easily missed. If not recognized and treated in a timely manner, brucellosis can lead to serious complications including death. The majority of cases in the United States are linked to consumption of unpasteurized dairy products imported from Mexico or travel to endemic areas. All health care providers should keep this infection in mind while dealing with patients presenting with fever. Clinicians should be vigilant and investigate for recent travel, consumption of unpasteurized dairy products, and animal exposure in these patients. More importantly, this pathogen can be used as a biological weapon as well. The article depicts a case of acute brucellosis in a patient who presented to an emergency room with a low-grade fever and nonspecific symptoms. Additionally, it reviews the crucial information regarding brucellosis and emphasizes important points on how to avoid misdiagnosis.

Introduction Patients often seek medical attention in an urgent manner when experiencing fever. An elevated body temperature is usually associated with an acute, common infectious illness. All clinicians are aware and familiar with how to diagnose and manage these cases. However, a rare infectious entity will occasionally be linked to those cases, particularly when the fever is of unknown origin (no clear source can be easily identified based on history and physical examination). Brucellosis is the most common zoonosis found in humans worldwide, particularly in endemic areas and in high-risk groups including animal handlers, slaughterhouse workers, veterinarians, laboratory workers, and

people consuming unusual foods. It is also called undulant fever because of its characteristic of recurrent febrile episodes. It is paramount that all health care providers in both endemic and non-endemic areas are aware of this febrile illness, as brucellosis is often misdiagnosed or overlooked. Untreated brucellosis leads to severe debilitation as well as high morbidity in patients of all ages. This article will present a patient case and review the crucial information regarding brucellosis.

Case Scenario RR, a 34-year-old male who is a welder, presents to the emergency department (ED) in a large city in Texas. He only speaks Spanish and has no significant past medical, surgical, social, or family history. Patient complains of headache, low back pain, fever occurring at random, and anorexia for three days. His review of systems is negative. He takes no medications.

Physical examination Vital Signs: Temperature 38.7°C, Pulse 96, Blood Pressure 122/71, Respiration Rate 20 General: Alert and oriented times three, no distress, well-developed, and nourished male. Head, Eyes, Ears, Nose, Throat: Normocephalic, pupils equally round and reactive to light and accommodation, extraocular muscles intact, normal tympanic membranes and pharynx. Neck: Supple, no adenopathy, masses, or jugular venous distension. Chest: Non-tender, lungs clear to auscultation, good air movement.

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American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

CV: Heart with regular rate and rhythm, no murmur or gallop, pulses equal and strong. Abdomen: Non-tender, soft, not distended, no organomegaly, bowel sounds normal. Extremities: No cyanosis, clubbing, or edema; capillary refill less than 2 seconds. Back: Normal inspection, no costovertebral angle tenderness or rebound. Neurological: Cranial nerves II-XII grossly intact, no focal deficits, normal gait, negative Romberg, Kerning’s, and Brudzinski’s signs. The initial workup included complete blood count, comprehensive metabolic panel, sedimentation rate, blood cultures, chest x-ray, computed tomography (CT) of the head, urine analysis, influenza screen, and cerebrospinal fluid (CSF) analysis. The results were remarkable for monocytosis on complete blood count, hyponatremia of 128, and sedimentation rate of 17 (mild elevation). Patient was treated with acetaminophen, intravenous fluids, and intravenous antibiotics in ED. His condition improved and he was discharged home with a prescription for levofloxacin and a recommendation to return if his symptoms deteriorate. Three days later, blood cultures revealed a growth for gram-negative rods, and further laboratory testing was positive for Brucella melitensis. The patient was called and prescribed doxycycline plus rifampin for six weeks.

Background Human brucellosis is a major bacterial zoonosis of global importance.1 In the United States, anywhere from 200 to 900 cases are reported annually,2 mostly due to Brucella melitensis.3 The disease is transmitted by inhalation, animal contact (cattle, sheep, goats, pigs, dogs), and consumption of unpasteurized dairy products and undercooked meat products.4,5 The vast majority of cases in the United States are linked to consumption of unpasteurized dairy products imported from Mexico.6 Of note, this pathogen can potentially be used as a biological weapon as well.7 Historically, the bacterium was first isolated in 1887 by David Bruce from the spleen tissue of a patient that had died as a result of undulant fever.1 However, organisms resembling brucellae have been detected in carbonized cheese from the Roman era.8 The bacterium is a small, non-motile, facultative, intracellular, and aerobic coccobacillus.7 Gram staining demonstrates tiny gram-negative rods lacking capsules, spores, and flagella.7 Bacterial growth is slow and requires special handling by a laboratory (need for 10% carbon dioxide). There are several isolated species of the bacterium. Brucella melitensis is the most common causative agent of human infection.6 The incubation period is usually between one and four weeks which is followed by flu-like symptoms characterized by an undulating fever pattern, weakness, malaise, headache, low back pain, rash, and fatigue.9 Physical examination is often

non-specific but may reveal hepatomegaly, splenomegaly, and/ or lymphadenopathy.7 Besides acute illness, brucellosis can be manifested as a focal process affecting practically any organ with osteomyelitis as most the common complication.10 In addition, some cases will progress into a chronic illness characterized by intermittent back pains, arthralgia, sweats, and psychosis with personality changes.11 Apart from high clinical suspicion, a clinician has several diagnostic tools to make a diagnosis of brucellosis. Diagnosis is predominantly based on serological assays and includes cultures, antigen detection by enzyme-linked immunosorbent assay (ELISA), genome detection using polymerase chain reaction (PCR), antibody detection, agglutination tests, and Coombs test.12 Unfortunately, the recognition of this zoonosis may be missed at presentation. As a result, there is often a delay in starting proper antibiotics and follow-up care. This is likely due to the fact that the symptoms are non-specific with few clues in the patient’s history. Currently, there is no vaccine available to prevent brucellosis in humans. Vaccines are available for cattle, sheep, and goats, but not pigs. The vaccines are live attenuated ones and they require careful handling as accidental exposure can lead to infection.13 Pasteurization of milk is also extremely important in the prevention of acquiring the disease by humans.

Discussion Our patient presented with non-specific symptoms suggesting an infectious process. Differential diagnosis includes upper respiratory infection (bacterial or viral), pneumonia, meningitis, or fever of unknown origin. He had a few symptoms and clinical clues that should have prompted us to consider brucellosis as part of the differential diagnosis list. One of them was low back pain that likely represented sacroiliac joint inflammation, a frequent finding in acute brucellosis.15,16,17 Non-specific symptoms and signs, a language barrier, a busy ED setting, and a well appearing patient did not prompt providers to look for other clues in the patient’s history that might reveal possible exposure to zoonotic infection. In fact, this patient was previously misdiagnosed and discharged with a diagnosis of a viral syndrome. The only reason he was properly diagnosed was by obtaining blood cultures. On the third day of incubation, the blood cultures revealed a growth of gram-negative small rods in aerobic bottles. They were fastidious organisms requiring a longer incubation time for growth. The identification was performed by PCR from the Centers for Disease Control and Prevention (CDC) confirming Brucella melitensis. Our case shows a classic scenario of a patient presenting with acute febrile illness to ED that was eventually diagnosed with the most common human zoonosis, namely brucellosis. However, it is an entity that is rarely seen in most clinical settings and therefore can be easily missed and mismanaged.11 Missing brucellosis or delay in diagnosis may be associated with several clinically significant consequences. Acute untreated brucellosis may lead to a chronic illness that can linger for years.11,18 These patients will develop a cyclic course characterized by sweats,

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arthralgias, back pain, and psychosis.19 Further, untreated brucellosis can lead to potentially life-threatening complications and subsequently death.10,13,18,20 These complications may be associated with the following system involvement: • osteoarticular such as inflammation of any joint; most commonly sacroiliac and sternoclavicular.15,16,17 • genitourinary such as orchitis, epididymitis, prostatitis, cystitis, nephritis, and glomerulonephritis.4,21 • pulmonary.10, 22 • gastrointestinal such as hepatitis, acalculous cholecystitis, pancreatitis, colitis, peritonitis, and abscesses.18 • hematological such as anemia, leukopenia, thrombocytopenia, pancytopenia, and disseminated intravascular coagulation (DIC).5,20 • neurological/psychiatric such as meningitis, encephalitis, myelitis, radiculitis, neuritis, behavior changes, and psychosis.4,13,18,20 • cardiac such as endocarditis, myocarditis, pericarditis, and aneurysm of the aorta or ventricles.10,13,18,20,23,24,25 • ocular such as uveitis, keratoconjunctivitis, corneal ulcers, optic neuritis, papilledema, and endophthalmitis.15,26 • dermatological such as skin ulcerations, petechiae, vasculitis, abscesses, and variety eruptions.10,20,27 Interestingly, patients with untreated acute brucellosis represent a potential occupational and environmental hazard to health care workers including laboratory workers who handle specimens without brucellosis precautions.28,29,30

Conclusion Brucellosis is diagnostically challenging and an uncommon disease in the United States, with Texas reporting approximately a third of cases.14 In the last decade, a new surge in brucellosis cases in animals has been detected in several states including Alaska, Montana, Wyoming, and Idaho.31,32 In Alaska, the surge might be explained by the impact of climate change which allows more animals to survive cold weather and subsequently increases the opportunity for transmission to humans. In addition, travel and exposure of military personnel during deployment to military conflicts in the Middle East and other endemic regions could potentially contribute to the increased number of brucellosis cases on US soil.33 Clinicians are encouraged to remember that brucellosis is an important zoonosis posing substantial health threats and morbidity in untreated cases. It is imperative to recognize the clinical symptomatology of brucellosis: fever of unknown origin, low back pain, myalgias, rash, arthralgia, or lymphadenopathy. Another key point is to obtain an exposure history (ask about potential exposures to animals, including pets, ingestion of unpasteurized milk or cheese, and a travel history). The understanding of local and endemic epidemiology is also crucial.

In addition, Brucella species are of interest as a biological weapon by various enemy groups and the military.7,34,35 In 1954, Brucella suis was first used as a biological weapon in the US.36 No other cases have been reported since then. The Armed Forces Health Surveillance Center (AFHSC), which is a part of the US Department of Defense, lists brucellosis as a potential biological agent.37 This organism offers several features that make it suitable for use as a bioarm: it is the most common zoonosis worldwide; airborne transmission is possible; it is highly contagious; it can enter the human body through conjunctivae, oropharynx, respiratory tract, and skin abrasions; only 10-100 organisms in aerosol are required to establish infection; and it is extremely difficult to diagnose.7,37,38,39 Hence, ED providers should have the ability to effectively triage those patients that might have been exposed to the infection as a result of a biological weapon.

Acknowledgement The author would like to thank Jennifer Skarpetowski for editing assistance. Potential Financial Conflicts of Interest: By AJCM® policy, all authors are required to disclose any and all commercial, financial, and other relationships in any way related to the subject of this article that might create any potential conflict of interest. The authors have stated that no such relationships exist.

Andrzej Skarpetowski, MD, is an emergency room physician at Baptist Emergency Hospital in San Antonio, TX.

References 1.

Cutler SJ, Whatmore AM, Commander NJ. Brucellosis-new aspects of an old disease. J Appl Microbiol. 2005;98(6):1270-81.

Dean AS, Crump L, Greter H, Schelling E, Zinsstag J. Global burden of human brucellosis: a systematic review of disease frequency. PLoS Negl Trop Dis. 2012;6(10):e1865.

Pappas G, Papadimitriou P, Akritidis N, Christou L, Tsianos EV. The new global map of human brucellosis. Lancet Infect Dis. 2006;6(2):91-9.

Bosilkovski M, Krteva L, Dimzova M, Kondova I. Brucellosis in 418 patients from Balkan Peninsula: exposure-related differences in clinical manifestations, laboratory results, and therapy outcome. Int J Infect Dis. 2007;11(4):342-7.

Bosilkovski M, Krteva L, Dimzova M, Vidinic I, Sopova Z, Spasovska K. Human brucellosis in Macedonia-10 years of clinical experience in endemic region. Croat Med J. 2010;51(4):327-36.

Levinson W, Jawetz E. Medical Microbiology & Immunology. Stamford, Conn: Appleton & Lange; 1996.

Pappas G, Panagopoulou P, Christou L, Akritidis N. Brucella as a biological weapon. Cell Mol Life Sci. 2006;63(19-20):2229-36.

Capasso L. Bacteria in two-millenia-old cheese, and related epizoonoses in Roman populations. J Infect. 2002;45(2):122-7.

Young EJ. An overview of human brucellosis. Clin Infect Dis. 1995;21(2):283-9.

10. Colmenero JD, Reguera JM, Martos F, at al. Complications associated with Brucella melitensis infection: A study of 530 cases. Medicine (Baltimore). 1996;75(4):195-211. 11. Young EJ. Brucellosis: current epidemiology, diagnosis, and management. Curr Clin Top Infect Dis. 1995;15:115-28.

Undulant Fever in ED: A Case Report

American Journal of Clinical Medicine® • Summer 2014 • Volume Ten Number Two

12. Christopher S, Umapathy BL, Ravikumar KL. Brucellosis: review on the recent trends in pathogenicity and laboratory diagnosis. J Lab Physicians. 2010;2(2):55-60. 13. Mantur BG, Amarnath SK, Shinde RS. Review of clinical and laboratory features of human brucellosis. Indian J Med Microbiol. 2007;25(3):188-202. 14. Glocowicz J, Stonecipher S, Schulte J. Maternal and congenital brucellosis in Texas: changing travel patterns and laboratory implications. J Immigr Minor Health. 2010;12(6):952-5.

35. Doganay GD, Doganay M. Brucella as a potential agent of bioterrorism. Recent Pat Antiinfect Drug Discov. 2013;8(1):27-33. 36. Guihot A, Bossi P, Bricaire F. [Bioterrorism with brucellosis]. Presse Med. 2004;33(2):119-22. 37. Burke RL, Kronmann KC, Daniels CC, et al. A review of zoonotic disease surveillance supported by the Armed Health Surveillance Center. Zoonoses Public Health. 2012;59(3):164-75.

15. Al-nakshabandi NA. The spectrum of imaging findings of brucellosis: a pictorial essay. Can Assoc Radiol J. 2012;63(1):5-11.

38. Bossi P, Tegnell A, Baka A, et al. Bichat guidelines for the clinical management of brucellosis and bioterrorism-related brucellosis. Euro Surveill. 2004;9(12):15-6.

16. Bosilkovski M, Krteva L, Caparoska S, Dimazora M. Osteoarticular involvement in brucellosis: study of 196 cases in the Republic of Macedonia. Croat Med J. 2004;45(6):727-33.

39. Greenfield RA, Drevets DA, Machado LJ, Voskuhl GW, Cornea P, Bronze MS. Bacterial pathogens as biological weapons and agents of bioterrorism. Am J Med Sci. 2002;323(6):299-315.

17. Geyik MF, Gür A, Nas K, et al. Musculoskeletal involvement of brucellosis in different age groups: a study of 195 cases. Swiss Med Wkly. 2002;132(7-8):98-105. 18. Gotuzzo E. Brucellosis. In: Tropical Infectious Diseases. Principles, Pathogenesis & Practice, Guerrant RL, Walker DH, Weller PF (Eds); Churchill Livingstone, Philadelphia. 1999. p. 498. 19. Spink WW. What is chronic brucellosis? Ann Inter Med. 1951;35(2):358-74. 20. Doganay M, Aygen B. Human brucellosis: an overview. Int J Infect Dis. 2003;7:173-82. 21. Akinci E, Bodur H, Cevik MA, et al. A complication of brucellosis: epididymoorchitis. Int J Infect Dis. 2006;10(2):171-7. 22. Pappas G, Bosilkovski M, Akritidis N, Mastora M, Krteva L, Tsianos E. Brucellosis and the respiratory system. Clin Infect Dis. 2003;37(7):e95-9. 23. Raju IT, Solanki R, Patnaik AN, Barik RC, Kumari NR, Gulati AS. Brucella endocarditis - a series of five case reports. Indian Heart J. 2013;65(1):72-7. 24. Jariwala P. 3D transthoracic echocardiography of Brucella endocaortitis and endocarditis of the aortic valve and ascending aorta. Echocardiography. 2013;30(7):E215-7. 25. Aksakal E, Sevimli S, Gürlertop Y, Tas H. An intracardiac mobile mass: ruptured left-ventricular false tendon with big vegetation due to Brucella endocarditis. Anadolu Kardiyol Derg. 2010;10(6):557-8. 26. Rolando I, Olarte L, Vilchez G, et al. Ocular manifestations associated with brucellosis: a 26-year experience in Peru. Clin Infect Dis. 2008;46(9):1338-45. 27. Ariza J, Servitje O, Pallare R, et al. Characteristic cutaneous lesions in patients with brucellosis. Arch Dermatol. 1989;125(3):380-3. 28. Fiori PL, Mastrandrea S, Rappelli P, Cappuccinelli P. Brucella abortus infection acquired in microbiology laboratories. J Clin Microbiol. 2000;38(5):2005-6.

EDITORIAL

Vijaya Arun Kumar, MD, FAAFP Emergency Medicine

This brucellosis case study gives a good description of the clinical presentation, diagnosis, and treatment modality for brucellosis, which is becoming more relevant in the age of biological warfare and increased international travel. Incidence of brucellosis is highly underestimated; a systematic analysis shows that relying on hospital data underestimates it 12-18 fold.1 A case study has its inherent drawbacks, but brucellosis is a difficult diagnosis to make clinically, and most case series show that the diagnosis is usually made once the patient is already admitted for fever of unknown origin.2,3 Brucellosis can be transmitted sexually4 and patients might not volunteer this information unless specifically asked. Also, the clinical presentations of human brucellosis can be vague and nonspecific. The most common symptom among these patients is subacute low back pain5, which can be easily overlooked. Blood culture is still the standard method and is often effective during the acute phase; the lysis concentration method gives the best results.6

References

29. Harding AL, Byers KB. Epidemiology of laboratory-associated infections. In: Biological safety: principles and practices (Third edition), Fleming DO, Hunt DL (Eds). ASM Press, Washington, DC. 2000. p.35.

30. Gruner E, Bernasconi E, Galeazzi RL, Buhl D, Heinzle R, Nadal D. Brucellosis: an occupational hazard for medical laboratory personnel. Report of five cases. Infection. 1994;22(1):33-6.

Dean AS, Crump L, Greter H, Schelling E, Zinsstag J. Global burden of human brucellosis: a systematic review of disease frequency. PLoS Negl Trop Dis. 2012;6(10):e1865.

31. Hueffer K, Parkinson AJ, Gerlach R, Berner J. Zoonotic infections in Alaska: disease prevalence, potential impact of climate change and recommended actions for earlier disease detection, research, prevention, and control. Int J Cirumpolar Health. 2013;72

Starakis I, Mazokopakis EE, Bassaris H. Unusual manifestations of brucellosis: a retrospective case series in a tertiary care Greek university hospital. East Mediterr Health J. 2010;16(4):365-70.

32. Cross PC, Maichak EJ, Brennan A, Scurlock BM, Henningsen J, Luikart G. An ecological perspective on Brucella abourtus in the western United States. Rev - Off Int Epizoot. 2013;32(1):79-87.

Kochar DK, Gupta BK, Gupta A, Kalla A, Nayak KC, Purohit SK. Hospital-based case series of 175 cases of serologically confirmed brucellosis in Bikaner. J Assoc Physicians India. 2007;55:271-5.

Wyatt HV. Brucella melitensis can be transmitted sexually. Lancet. 1996;348(9027):615.

33. Bechtol D, Carpenter LR, Mosites E, Smalley D, Dunn JR. Brucella melitensis infection following military duty in Iraq. Zoonoses Public Health. 2011;58(7):489-92.

Geyik MF, Gür A, Nas K, et al. Musculoskeletal involvement of brucellosis in different age groups: a study of 195 cases. Swiss Med Wkly. 2002;132(7-8):98-105.

34. Anderson PD, Bokor G. Bioterrorism: pathogens as weapons. J Pharm Pract. 2012;25(5):521-9.

Matar GM, Khneisser IA, Abdelnoor AM. Rapid laboratory confirmation of human brucellosis by PCR analysis of a target sequence on the 31-kilodalton Brucella antigen DNA. J Clin Microbiol. 1996;34(2):477-8.

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