PE Poster Presentation-Endocrine (1-35

PE01

A WOMAN WITH STIFF-PERSON SYNDROME—AUTOIMMUNE POLYENDOCRINE SYNDROME TYPE 2

1MEI-CHEN YEH, 1NAI-CHENG YEH, 1SHANG-GYU LEE, 1KAI-JEN TIEN,

1CHEWN-YI YANG

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Chimei medical center, Taiwan, R.O.C.

A 41-year-old woman was seen in the emergency department(ED) at this hospital because of trunkal spasm and rigidity.

The patient had been well until approximately 1 week before this ED visit, when spasms and rigidity developed in her trunk and neck. The muscle spasms and rigidity progressively worsened. One walking. She came to the emergency department at this hospital.

At ED, her serum glucose was 476mg/dL, and hyperglycemic chorea was the impression. Her serum ketone body was 0.8mmol/L and blood osmolality was 310 mOsm/kg. She was given 6 unit of insulin subcutaneously, normal saline hydration and diazepam intravenously, and then she was discharged from ED with the prescription of oral-form antidiabetic medicine, Metformin(500mg) twice daily after meal. And she was refer to endocrine outpatient clinic 2 days later.

At endocrine outpatient clinic, her blood glucose was 230mg/dL, however, the spasms and rigidity in trunk and neck still existed. Hyperglycemic chorea is not likely. This patient mentioned that she was told to have suspected hyperglycemia during her pregnancy more than 10 years ago, and she was also told that her blood sugar returned to normal range after her pregnancy. She denied any other systemic disease except for the suspected hyperglycemia.

She denied allergic to any medicine. She did not smoke, drink alcohol, or use illicit drugs. She denied family history of any systemic disease.

On examination, the vital signs were normal, except for elevated heart rate. The abdomen was rigid due to muscle spasm without tenderness on palpation. Mild stared eyes were seen. The remainder of the examination was normal. The hematocrit, hemoglobin level, platelet count, creatinine, and GPT were normal. Other test were as follows: Hba1c 10.6 %, GAD-65 > 2000 U/mL. Thyroid hormones were check because mild stared eyes were noted. (T4: 9.2ug/dl, TSH 0.03uIU/ml, free T4 1.77ng/dL, anti-TPO 190.9IU/mL).

Thyroid sonography showed mild enlargement of bilateral lobes of thyroid gland with mild increased vascularity. I131 uptake scan was performed and the thyroid uptake at 24 hour was 80%(Normal range 15%-25%), consistent with diffuse thyrotoxicosis.

Glucagon test for suspected type 1 diabetes was performed and the result revealed C-peptide at

0 minute 0.65 ng/mL, at 6th minute 1.07 ng/mL, which is compatible with type 1 diabetes mellitus. Neurologist was consulted and stiff-person syndrome is highly suspected.

The current impression is:

Autoimmune polyendocrine syndrome type 2 (Type 1 diabetes mellitus and Graves’ disease)

Stiff-person syndrome The current treatment is basal bolus insulin for diabetes mellitus and oral form diazepam for muscle spasm and stiffness.

PE02

A CASE REPORT : ANTITHYROID DRUG-INDUCED AGRANULOCYTOSIS IN A 55 YEAR-OLD WOMAN

1YAN-YU LIN, 1,2CHUNG-HUEI HSU, 1CHEN LING HUANG, 1SHUEN-FU WENG,

1YU-PEI LIN

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Taipei Medical University Hospital, Taipei, Taiwan; 2Department of Nuclear Medicine, Taipei Medical University Hospital, Taipei, Taiwan

Introduction: Antithyroid drugs are used commonly in hyperthyroidism patients, which includes propylthiouracil , carbimazole and its active metabolite methimazole. Most patients have several ATD-induced agranulocytosis is rare, but the severity of this possibly condition could be lifethreatening. Earlier diagnosis and the appropriated managements are very important.

Case Report: This 55-year-old woman has history of hyperthyrodism under Methimazole(5 mg) 1 tab TID treatment. After three months later, fever with sore throat was noted that she came to ER for help. In addition, right-lower quadrant abdominal dull pain was also occurred. Initial lab data revealed that: WBC 860/ul, Seg 2.0% (ANC 17.2), CRP 14.13mg/dl. Influenza A, B were all negative. Abdomen CT revealed prominent appendix with relative mild thickening wall (Fig. 1). Due to neutropenia, oncologist was also consulted and peripheral blood smear was performed. No blast cell nor promyelocyte were seen. Under the impression of Antithyroid drug-Induced agranulocytosis , we stopped methimazole use and the patient was admitted for observation with empiric antibiotics stabilized WBC/DC was noted, and all parameters were within normal range after 1 week follow up. No fever noted and right-lower quadrant abdominal dull pain was also subsided then. The patient was arranged radioactive iodine then for restore an euthyroid state.

Discussion: Agranulocytosis occurs in only 0.2–0.5% of patients with Graves’ disease receiving antithyroid drugs. The rarity of this side effect often makes our clinical physicians miss it and probably let patients life-threatening. High fever and sore throat are the most common presenting signs, but patients may also be asymptomatic. Genome-wide association studies in ethnic Chinese people in Taiwan and Hong Kong have shown an association between agranulocytosis induced by antithyroid drugs and the HLA alleles HLA-B*38:02 and HLA-DRB1*08:03. Treatment requires immediate stop of the antithyroid drug and initiation of broad-spectrum antibiotics. Hematopoietic growth factors may be used. Early diagnosis and treatment can prevent the patient from catastrophic event of ATD-induced agranulocytosis.

PE03

COEXISTENCE OF RESISTANCE TO THYROID HORMONE SYNDROME AND PITUITARY GRANULAR CELL TUMOR IN A TAIWANESE YOUNG LADY, MIMICKING TSH SECRETING TUMOR

1,2YU-YI LIN, 5CHI-LUNG TSENG, 1,3,4CHUN-JUI HUANG

1Division of Endocrinology and Metabolism, Department of Medicine,Taipei Veterans General Hospital, Taiwan 2Department of Medicine, Cheng Hsin General Hospital, Taipei, Taiwan; 3Faculty of Medicine, School of Medicine, National Yang-Ming University, Taipei, Taiwan; 4Institute of Public Health, National Yang-Ming University, Taipei, Taiwan; 5Department of Internal Medicine,Taoyuan General Hospital,Ministry of Health and Welfare,Taipei,Taiwan

Background: Resistance to thyroid hormone (RTH) syndrome is a rare disorder which presents as high free thyroid hormones with unsuppressed thyroid-stimulating hormone (TSH) secretion and diminished responsiveness to thyroid hormones by target organ. Appropriate diagnosis is crucial to avoid over-treatment with anti-thyroid drugs, radio-iodine ablation, and/or pituitary surgery.

Method: tumor but later proved to have pituitary granular cell tumor instead.

Case presentation: A 33-year-old Taiwanese married woman was found to have elevated free T4: for evaluation of palpitation and difficult conception in 2013. Besides mild tachycardia (heart rate: 98 beats per minute), she had no other symptoms and signs of hyperthyroidism. Propylthiouricil was prescribed by her gynecologist and discontinued one year later due to persistence of abnormal thyroid hormone levels under medication. Afterwards, she was referred to an endocrinologist and was told to have central hyperthyroidism based on negative thyroid autoantibodies, moderately enlarged homogenous thyroid glands on thyroid sonography, and a 1.2 cm right pituitary adenoma on sella magnetic resonance imaging (MRI). Endoscopic trans-phenoidal surgery for pituitary tumor removal was performed in May, 2017. Histopathology revealed granular cell tumors that were positive for S-100 protein and TTF-1 but negative for TSH, ACTH, GH and prolactin on immunohistochemical staining. In February 2018, she visited Taipei Veterans General Hospital due to persistence of abnormal thyroid hormone levels (free T4: 2.19 ng/dL; T3: 209 ng/dL, normal: 58-159; total T4 showed no residual pituitary tumor. After administration of 200 mcg of thyrotropin releasing hormone (TRH) intravenously, exaggerated TSH response was noted, with TSH levels showing 6.0, 24.7, 22.6, planned, Inderal 5mg thrice daily was used to lower T3 levels in case if a non-RTH fetus exists.

Conclusion: Although rare, RTH should be considered in the differential diagnosis of high thyroid hormone levels with unsuppressed TSH. The presence of a pituitary adenoma does not conclude a TSH secreting tumor, like our case. We suggest careful evaluation with functional and genetic testing prior to initiation of therapy in order to avoid unnecessary surgeries and medications. A favorable outcome generally follows a diagnosis of RTH. Special consideration needs to be taken in mothers with RTH harboring wild-type fetus.

PE04

A RARE CASE OF ECTOPIC ADRENOCORTICOTROPIC HORMONE (ACTH) SYNDROME DUE TO THYMIC LARGE CELL NEUROENDOCRINE CARCINOMA PRESENTING AS SEVERE HYPOKALEMIA IN A TAIWANESE MAN

1,2YU-YI LIN, 1,3LIANG-YU LIN

1Division of Endocrinology and Metabolism, Department of Medicine,Taipei Veterans General Hospital, Taiwan 2Department of Medicine, Cheng Hsin General Hospital, Taipei, Taiwan 3Faculty of Medicine, School of Medicine, National Yang-Ming University, Taipei, Taiwan

Background: Ectopic ACTH syndrome (EAS) represented 5% of Cushing syndrome. EAS is rapid onset, severe, associated with hypercortisolism related life-threatening infections and hypokalemia. In 60% of cases, the tumor is located in intrathoracic organ (lung, bronchus and thymus). Large cell neuroendocrine tumor (LCNEC) of thymus secreting ACTH is a high grade, aggressive, poor prognosis and only few case reports in literature.

Method: We report a case of thymic LCNEC related EAS, which presented as severe hypokalemia, metabolic alkalosis and muscle weakness.

Case presentation: A 71-year-old man presented with 12-kg weight loss, progressive generalized weakness and bilateral legs edema for two months.He has medical histories of hypertension and diabetes mellitus. Physical examination revealed generalized weakness without Cushingoid appearance. Laboratory examination showed metabolic alkalosis and severe hypokalemia (1.7 mmol/ L, range: 3.4-4.7).Hypokalemia work-up showed high morning ACTH 497 pg/mL (normal: < 46.0), survey showed lack of ACTH/cortisol circadian rhythm, markedly increased 24h urinary free cortisol suppression tests showed non-suppressible. Ovine corticotrophin-releasing hormone stimulation test and desmopressin (DDAVP) test showed ectopic ACTH source. Pituitary MRI revealed negative Ectopic ACTH syndrome was impressed. Etomidate continuous infusion was prescribed.Fournier gangrene of left scrotum occurred during hospitalization and antibiotic and surgical treatment were prevascular of mediastium. In-111 Octreoscan revealed several nodular lesions with increased In-111 uptake in the paraortic and prevascular region considering neuroendocrine tumor. Anterior mediastinal tumor excision was performed and histopathological report showed large cell neuroendocrine carcinoma of thymus with mitotic count 13/10 HPF and positive immunohistochemical stain for synaptophysin, chromogranin, DD-56, SSTR2 and ACTH. Sandostain LAR injection was administered

afterward. Unfortunately, tumor progression with hypercortisolemia occurred after 6 months . External radiation and systemic chemotherapy were performed later.

Conclusion: Although rare, Cushing syndrome must be considered in differential diagnosis of hypokalemia like our case, a challenging case of EAS. Early diagnosis and treatment was important to avoid life-threatening infections and metabolic complications. Moreover recurrence free survival with Sandostain LAR alone was only 6 months for LCNEC.

PE05

PARADOXICAL RESPONSES TO DYNAMIC TESTING FOR CUSHING’S DISEASE: AN UNUSUAL CASE OF LARGE CORTICOTROPH ADENOMA WITH EXTENSIVE LOCAL INVASION

1HSUAN-WEI LIN, 1SHEAU-FANG PAN, 1YU-YI LIN, 1FANG-YU CHEN, 1,2CHII-MIN HWU

1Division of Endocrinology and Metabolism, Department of Medicine, Taipei Veterans General Hospital, Taipei, Taiwan, R.O.C.; 2School of Medicine, National Yang-Ming University, Taipei, Taiwan

Background: Cushing’s disease is most commonly caused by a pituitary microadenoma secreting excessive ACTH. Biochemical responses of corticotroph macroadenoma to dynamic testing are less described in the literature. Here we present the case of a patient with a large and invasive corticotroph adenoma causing Cushing’s disease.

Case: A 67-year-old woman presented to the emergency department with fever and neck swelling. She was diagnosed with deep neck infection in another hospital and was transferred to our hospital due biopsy of the intranasal tumor was done along with wound debridement for the deep neck infection. The pathology of the tumor was compatible with invasive corticotroph adenoma. Dynamic testing revealed cortisol secretion non-suppressible by high dose dexamethasone and not stimulated by ovine corticorelin administration, whereas desmopressin stimulation test was positive. Before definitive surgical treatment of the large adenoma, adequate control of hypercortisolism was achieved with etomidate infusion.

Conclusion: Cushing disease caused by pituitary macroadenomas has biochemical responses distinct from typical one with microadenomas. Such paradoxical diagnostic gap deserves attention to avoid confusion with ectopic ACTH syndrome.

PE06

OSTEOPOROSIS IN ADRENAL CUSHING’S SYNDROME: 2 CASE REPORTS

YU-LING LIN

Division of Endocrinology and Metabolism, Department of Internal Medicine, Feng yuan hospital, ministry of health and wealfare, Taiwan, R.O.C.

Background: Osteoporosis is one of the most common complication of Cushing syndrome. Besides, the reversible nature of osteoporosis is generally accepted, and normalization of bone mineral density following cure of Cushing syndrome could be achieved. Here we report 2 cases of adrenal Cushing’s syndrome and focus on bony complication.

Case 1: A 29-year-old female was referred to the endocrinology OPD due to obesity. PE revealed moon face, but no facial plethora, no acne, no central obesity, easily ecchymosis, increased hair over pre-auricular area and mild hypertension.

Lab: loss diurnal rhythm cortisol 8am/11pm 16.53/16.14 ug/dl; 1mg dexamethasone suppression test: 21(+), with low ACTH: <5/<5; Standard two-day 2 mg test: cortisol 16.55 ug/dl; Na/K: 137/3.7; PA/PRA: corrected ratio 9.8, VMA: 5.78mg/24hrs.

CT: adrenal tumor 28mm(Lt).

Pre-op BMD, spine(9908): T score -0.8, Z score -2.3

Post-op BMD, spine(10302): T score 0.9, Z score 0.0

Then patient received unilateral adrenectomy. Postop F/U 1mg DST: cortisol 0.59 ug/dl. Normal bone mineral density was noted following cure of Cushing’s syndrome

Case 2: A 50 y/o male was admitted due to falling down injury with lower legs weakness. Multiple old rib fractures & T-L spine compression fracture were noted. Past history of Lt adrenal tumor has been noted for yrs but patient refused surgical intervention; DM, HTN F/U at medical center. PE revealed moon face, easily bruise, paper skin.

Lab study: loss diurnal rhythm cortisol 8am/11pm 25.62/29.74 ug/dl;1mg dexamethasone suppression test: 27.60 ug/dl;low ACTH: 5.5/6.0 pg/ml; Na/K: 141/3.4, Ca: 9.1 mg/dl; PA/PRA: corrected ratio 14.79, VMA: 2.59 mg/24hrs(WNL) region, a left adrenal tumor /c lung and liver metastasis is considered.

L spine: multiple compression fracture of the T-L vertebral body- T11, L2, and L4.

BMD, Lt femoral neck, T score -3.1, Z score -2.5;

BMD, Rt femoral neck, T score -3.0, Z socre -2.3

Conclusion: Cushing syndrome comprised the S/S associated with prolonged exposure to inappropriately elevated levels of glucocorticoid. Glucocorticoids inhibit osteoblast function which

account for the osteopenia and osteoporosis. Besides, glucocorticoids also induce negative calcium balance by inhibiting intestinal calcium absorption and increasing renal calcium excretion and

PE07

A CASE REPORT OF SEVERE HYPOTHYROIDISM PRESENTING WITH ACUTE CONSCIOUS CHANGE AND SEIZURES

JUI-HSIANG LI, YOUCHENG CHEN, SU-HUEY LO

Department of Internal Medicine Tao-Yuan General Hospital

Brief history: A 38-year-old female had sudden onset of agitation with attack tendency. She was sent to our ER for evaluation and repeated generalized tonic-clonic seizure attack was observed duration about 30 seconds frequently. She was admission and seizure attack was still noted. Though, the EEG showed normal cortical function. The brain MRI revealed post-ictal right hemispheric brain swelling. The laboratory showed TSH = 214,Free T4 = 0.23 compatible with primary Hypothyroidism.

Discussion: Thyroid hormones act in the brain where they play an essential role in fetal and post-natal brain development as well as the maintenance of adult brain function. In addition, thyroid hormone promotes CNS repair, as it has been demonstrated that CNS re-myelination is dependent on these hormones. Hypothyroidism is insufficiency of thyroid gland to produce thyroid hormone may lead to cerebral hypoxia and predispose to confusion attacks and syncope. Psychiatric disorders are common and are usually of paranoid or depressive type and may induce agitation (myxedema madness). Epileptic seizures have been reported and tend to occur in myxedema crisis. The cause of seizure activity in hypothyroidism is unknown. It may be due to cerebral edema secondary to expansion of extracellular fluid volume. Inappropriate antidiurectic hormone(ADH) secretion, hyponatremia and hypoventilation with postanoxic encephalopathy maybe relate to this condition and further precipitate seizure activity. Management of myxedema crisis involves replacement of thyroid hormone with correcting predisposing factor and supportive care. Prior to thyroxine therapy, increasing cortisol clearance.

PE08

THYROID STORM WITH ECMO SUPPORT IN A 41-YEAR-OLD WOMAN: A CASE REPORT

1HSIN-WEI WANG,

1SHIH-CHANG LO, 1EDY KORNELIUS, 1YI-SUN YANG, 1CHIEN-NING HUANG

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Chung Shan Medical University Hospital, Taichung, Taiwan, R.O.C

Introduction: Thyroid storm is a life-threatening condition which have an exaggeration of the usual symptoms of hyperthyroidism. The clinical presentation includes fever, tachycardia, hypertension, and neurological and GI abnormalities.

Rapid diagnosis is important, because cardiogenic shock or multiple organ failure may occur if left untreated in some patients. Extracorporeal membrane oxygenation (ECMO) provide the last chance in patient with critical patients.

Case: A 41-year-old female presented with progressive shortness of breath for 2 days. She had underlying disease of Graves’ disease under regular medication control. She hold the anti-thyroid drug by herself since 2 weeks ago. Then progressive dyspnea, poor appetite, and diarrhea occurred. acute pulmonary edema were noted. Intubation was arranged due to hypoxemic respiratory failure. Propylthiouracil (PTU), Lugol’s solution, and systemic steroid were administered. However, refractory hypotension developed despite aggressive fluid challenge and maximal dose of vessel-constriction agent. Thyroid storm with acute heart failure and cardiogenic shock were suspected. After discussion with her family, ECMO was used. CVVHD was also arranged due to acute kidney injury with oligouria. Fortunately, the vital sign became stable gradually, and ECMO was removed followed by mechanical ventilation weaning successfully.

PE09

LONG-TERM OUTCOME AND PROGNOSTIC FACTORS OF SINGLE DOSE RADIOIODINE THERAPY IN PATIENTS WITH GRAVES’ DISEASE

1YI-TING YANG,

1PEI-WEN WANG, 1JUNG-FU CHEN, 1SHIH-CHEN TUNG, 1MING-CHUN KUO, 1SHAO-WEN WENG, 1CHEN-KAI CHOU, 1FENG-CHIH SHEN, 1CHIH-MIN CHANG, 1CHIA-JEN TSAI, 1CHENG-FENG TSAO

1Division of Metabolism, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan.

Background: There are few domestic studies regarding the long-term outcome of radioiodine(RAI) therapy in patients with Graves’ disease(GD). The aim of this study is to evaluate patients with GD.

Methods: This is a retrospective clinical study. There were 424 patients who received RAI treatment for hyperthyroidism between 1989 and 2016 in Kaohsiung Chang Gung Memorial Hospital. Excluding those with toxic adenoma, loss of follow-up, thyroidectomy or 2nd dose of RAI within 3 years after RAI therapy and whose charts were unavailable for review, totally 182 patients with GD thyroid antibodies. Kaplan-Meier curve, cox-regression model and ROC curve were used for statistical analysis.

Results: Loss of regular follow-up was found in 31.1% (108/347) of the patients. Among the 182 hyperthyroidism after thyroidectomy or prolonged drug therapy, 29 suffered from severe adverse antithyroid drug reaction. Most (75.8%) were females, and the mean age was 46.4 ± 13.8years. The mean RAI dose was 8.2 ± 3.3mCi(range 3-20mCi). The mean follow-up period was 108.8 ± 79.3 success and failure groups. Patients with grade 0-2 goiter had higher successful rate than patients with grade 3 goiter (p value = 0.002,HR = 2.02, 95%CI = 1.28-3.19). But if an early regression of grade 3 goiter to grade 0-2 in 3 months after RAI therapy, the successful rate was not inferior to those with initial grade 0-2 goiter. ROC curve revealed that the best timing to evaluate treatment result and the overall cut-off point was 27.5 months, while that for patients with grade 0-2 and grade 3 goiters was 11 months and 39months, respectively.

Conclusions: choice of therapy. Further, regular follow-up after RAI therapy should be emphasized.

PE10

NIVOLUMAB INDUCED THYROIDITIS IN PATIENT WITH HEPATOCELLULAR CARCINOMA - A CASE REPORT

1CHIH-CHEN WANG, 1HAO-CHANG HUNG, 1JING-HAN LIN, 1CHUN-CHUNG LIN, 1HORNG-YIH OU

1Division of Endocrinology and Metabolism, Department of Internal Medicine, National Cheng Kung University Hospital

A 59-year-old man initially visited our outpatient department because of sudden onset of right upper quadrant abdominal pain, and abdominal CT showed bilateral hepatic tumors. Liver biopsy confirmed the diagnosis of hepatocellular carcinoma. Since then, the patient received seven times of treatment course of TACE with Doxorubicin, but MRI still showed liver tumor recurrence. We then started HAIC with Cisplatin and Doxorubicin for three times. However, abdominal MRI still revealed progression of residual liver tumors. Due to poor response of treatment, we started Nivolumab therapy(3 mg/kg). After two courses, the patient reported tachycardia, and new onset of paroxysmal atrial fibrillation with rapid ventricular response was found. Thyroid function tests showed TSH: < 0.03 uU/ml and Free T4: 2.98 ng/dl. Thyroid autoimmunity showed microsomal Ab: positive, thyroglobulin Ab: positive, anti-mitochondrial Ab: 1:10(-) and Anti-TSH receptor: < 0.3(U/ L). Therefore, we empirically prescribed Methimazole 5 mg b.i.d by mouth since 2018/10/19 for suspicious thyrotoxicosis. After 1 week of the treatment, palpiatation was improved and laboratory data on 2018/10/24 showed Free T4: 1.72 (ng/dl)/T3: 103.75 (ng/dl). We adjusted Methimazole to 5mg q.d and kept it for one more month. Laboratory data on 2018/11/13 showed: TSH: 33.05(uU/ml)/Free

PE11

LENVATINIB AS SALVAGE TREATMENT IN RAI-REFRACTORY DTC PATIENTS WITH PRIOR TYROSINE KINASE INHIBITOR FAILURE: A SMALL CASE SERIES REPORT

1DANIEL HY SHEN,

2MING-LANG SHIH, 3REN-HUA YEH, 4SHAO-CHENG LIU, 1LI-FAN LIN, 1CHENG-HAN HO, 1YUEH LEE, 1CHENG-YI CHENG

1Department of Nuclear Medicine and PET Center, Tri-Service General Hospital, Taiwan, R.O.C.;

2Department of Surgery, Tri-Service General Hospital, Taiwan, R.O.C.; 3Department of Internal Medicine, Tri-Service General Hospital, Taiwan, R.O.C; 4Department of Head & Neck Surgery, Tri-Service General Hospital, Taiwan, R.O.C

Backgrounds: When differentiate thyroid cancer (DTC) becomes RAI-refractory (RAI-R), tyrosine kinase inhibitors (TKIs) such as Sorafenib and Lenvatinib might be useful to control the disease. On the basis of two phase III clinical trials (DECISION and SELECT) showing good therapeutic response and enhanced progression-free survival of both TKIs, both agents have been approved for treatment of RAI-R DTCs but, indeed, the earlier emergence of Sorafenib renders most such patients probably receive seems one of the choices for subsequent management in such patients with Sorafenib failure. Our study aims to investigate the usefulness of Lenvatinib as salvage treatment.

Methods: We retrospectively evaluated patients (N = 15) with RAI-R DTCs ever treated with Sorafenib first and then switched to Lenvatinib due to tumor progression in our institute. Tumor response was assessed by RECIST criteria, change of serum thyroglobulin (Tg) after treatment, and change of FDG avidity in terms of SUVmax of main tumor lesion(s) were measured. The frequency of drug-related adverse effects (AEs) graded by CTCAE v.4 were also analyzed.

Results: Twelve (80%) of 15 RAI-R DTC patients with Sorafenib failure showed good response to Lenvatinib treatment including stable disease (n = 3) and partial response (n = 9) with enduring response of median of 12 months. There is a dramatic drop of serum Tg in Lenvatinib responders on initial intervention showing -45 to -98% of change during 3-6 months after treatment. There is a marked change of lesional FDG avidity with -53% to -83% of change after at least 10 months of treatment. The drug-related AEs were recorded from all these RAI-R DTC patients (N = 15) including hypertension (n = 9), proteinuria (n = 6), fatigue (n = 6), diarrhea (n = 5), dermatitis or hair loss (n = 5) and sporadic ones like stomatitis, hoarseness, hypocalcemia, acute cholecystitis (n = 1, death), tumor bleeding (n = 1, death), open wound with necrosis (n = 1, death). Also some serum biochemical abnormalities like elevated TSH, AST, ALT, creatinine or amylase level were noted occasionally.

Conclusion: From our study, the use of Lenvatinib as salvage treatment for RAI-R DTC cases with Sorafenib failure are manifested by greatly reduced serum Tg and tumor FDG uptake. However, the management of common AEs as well as to prevent fetal AEs is needed. For those who show proteinuria

or serum biochemical abnormalities, further study to understand the long-term effect is warranted.

PE12

SUBACUTE THYROIDITIS WITH HYPOPARATHYROIDISM - A CASE REPORT

1SHU-HSUAN HUANG, 2HAO-CHANG HUNG, 2YE-FONG DU, 2HORNG-YIH OU

1Department of Internal Medicine, National Cheng Kung University Hospital, 2Division of Endocrinology and Metabolism, National Cheng Kung University Hospital

Subacute thyroiditis is usually a self-limited disease without complications, and hypothyroidism becomes permanent in rare cases. We report a case of subacute thyroiditis with hypoparathyroidism.

A 38-year-old woman presented with recurrent epigastric pain followed by muscle cramps and carpopedal spasms, together with chest tightness and anxiety. Laboratory tests showed severe hypocalcemia, hyperphosphatemia, hypomagnesemia, and low-normal iPTH level. These presentations were preceded by an episode of fever with upper respiratory tract symptoms one month ago. Thyroid function tests showed thyrotoxicosis with positive anti-thyroid peroxidase antibody and antithyroglobulin Ab. Thyroid ultrasonography demonstrated bilateral thyroid nodules less than 1 cm, and the I131 thyroid uptake/scan suspected subacute thyroiditis. Her symptoms subsided after correction of serum calcium level. She received calcium carbonate, calcitriol for primary hypoparathyroidism, and thyroxine support for hypothyroid phase one year later.

PE13

CRIBRIFORM-MORULAR VARIANT OF PAPILLARY THYROID CARCINOMA: A CASE REPORT AND REVIEW OF THE LITERATURE

1YUNG-NIEN CHEN, 2SHUN-CHEN HUANG, 1JUNG-FU CHEN, 1CHEN-KAI CHOU

1Division of Metabolism, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan, R.O.C.; 2Department of Pathology, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan, R.O.C.

Background: Cribriform-morular variant of papillary thyroid carcinoma (CMV-PTC) is a rare but distinct histological subtype of papillary thyroid carcinoma (PTC) associated with familial adenomatous polyposis(FAP). FAP is an autosomal dominant cancer predisposition syndrome characterized by the progressive development of multiple colorectal adenomatous polyps and an increased risk of colorectal carcinoma.

Methods: cancer histologically diagnosed with CMV-PTC and colon polyps.

Results: A 19-year-old girl presented to the endocrine clinic with palpable thyroid nodules of six months duration. Both of her grandmothers and two aunts had unknown thyroid disease. Fineneedle aspiration of thyroid was compatible with Bethesda category V: suspicious for malignancy. Because of her family history of FAP, colonoscopy was arranged and revealed large bowel polyposis. She underwent total thyroidectomy which revealed CMV-PTC. Histology was characterized by a prominent cribriform pattern of growth with interspersed cell clustered arranged as morules. Immunohistochemical (IHC) staining for CMV-PTC cells reveal beta-catenin in both the nuclei and cytoplasm, which is the key feature of this subtype.

Conclusion: Patients with CMV-PTC have distinctive clinical, pathological pattern and elevated risk of colorectal cancer when compared to conventional papillary thyroid carcinoma. Young thyroid cancer patients with strong family history of thyroid or colorectal cancer should be evaluated the possibility of CMV-PTC by IHC staining of beta-catenin. Due to its strong connection with FAP syndrome, further colonoscopy and genetic study of the APC gene sequencing should be also considered.

Keywords: Papillary thyroid carcinoma, Cribriform morular variant, Familial adenoatous polyposis

PE14

DOUBLE PRIMARY CANCERS IN PATIENTS WITH THYROID CANCER: A RETROSPECTIVE ANALYSIS REPORT IN SOUTHERN TAIWAN

1SHU-TING WU, 2SHUN-YU CHI, 1JUNG-FU CHEN, 1CHEN-KAI CHOU

1Division of Metabolism, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan, R.O.C. ; 2Division of General Surgery, Department of Surgery, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan, R.O.C.

Background: Thyroid cancer is the most common endocrine cancer. Thyroid cancer survivors had an increasing risk of double primary cancers. Patient with double primary cancers in the setting of thyroid cancer had worse prognosis. The aim of this study was to investigate the clinicopathological characteristics, sex differences in cancer incidence trends, and survival of these double primary such as radioactive iodine treatment for thyroid cancer and radiation therapy for breast cancer.

Methods: Between 2013 and 2017, a total of 935 thyroid cancer patients who had long-term follow-up at Kaohsiung Chang Gung Memorial Hospital in Taiwan were retrospectively reviewed. Clinical data were obtained from the medical records. Those with double primary cancers were

Results: A total of 84 patients with double primary cancers in the setting of thyroid carcinoma were identified from the 935 thyroid cancer patients, giving a prevalence rate of 9% at our center. The mean age of diagnosis with double primary cancers was 58.3 ± 10.6 years. There was a female preponderance (26 males and 58 females). The most common site of second primary cancer was the head and neck in men and the breast in women. Of the 84 double primary cancers, 26 were synchronous in nature and 58 were metachronous. Among synchronous malignancies (26/44 patients), H&N tumors were the most commonly associated. In further analysis, 12 patients had incidental

Conclusions: There has been an increase in the number of patients diagnosed with double primary cancers with thyroid cancer. This trend can be due to prolonged life span, the increase survival rate of cancer patients, and better diagnostic techniques. The relationship between the double primary cancers has been investigated but the exact attributive factors and pathophysiology still controversial. Greater awareness of the increasing prevalence of multiple primary cancers in our clinical practice is required among both cancer patients and clinical physicians.

PE15

CLINICAL EXPERIENCE OF TSHOMA IN CHANG-GUNG MEMORIAL HOSPITAL

1YU-LING LU, 1MIAO-JHEN LIOU, 1CHENG-WEI LIN, 1SZU-TAH CHEN

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Chang Gung Memorial Hospital, Chang Gung University, Taiwan, R.O.C.

Background: Thyroid-stimulating hormone secreting pituitary adenoma (TSHoma) is a rare cause of of hyperthyroidism. Here we report 3 cases of TSHoma diagnosed and treated in a referral medical center.

Methods: With the finding of an abnormally elevated TSH level, 3 cases of TSHoma were of 3 TSHoma cases were retrospectively analyzed.

Results: The demographic characteristics of 3 TSHomas were illustrated in Tab. 1. All the 3 patients had high free T4 (fT4) and normal to high TSH levels initially. Two of them received TRH stimulation test with blunted TSH response. For suspected TSH secreting pituitary tumor, all patients underwent sella MRI/CT, and pituitary macro- or micro-adenoma were disclosed with positive stain for multi-hormones. All patients received trans-sphenoidal surgery (TSS) to resect/biopsy of the pituitary adenoma. The situation after TSS was different, hypothyroid developed initially and returned to euthyroid in case 1 with residual tiny tumor. High fT4 and normal TSH level initially, then became euthyroid and adrenal insufficiency in case 2 with residual macroadenoma. Persistent high fT4 with low-normal TSH was found in case 3 although sella MRI showed absence of residual tumor. Euthyroidism was achieved after bromocriptine (BMC) treatment, and the patient eventually received

Conclusions: TSHoma should be taken into consideration in hyperthyroid patients with normal to high TSH. TRH stimulation test may not differentiate primary from secondary hyperthyroidism and/or resistance to thyroid hormone syndrome. The tumor size was correlated with time lag to a definite diagnosis of TSHoma, but not correlated with the use of anti-thyroid drug (ATD). In the two macroadenomas (cases 1 and 2) with growth hormone (GH) and prolactin (PRL) co-secretion, biochemical remission was achieved even with postoperative residual tumors. In the folliclestimulating hormone (FSH) and luteinizing hormone (LH) co-secreting microadenoma (case 3), persistently elevated TSH was found postoperatively without radiological evidence of residual tumor (although a response to BMC was observed). Thus, the clinical course and outcome of TSHomas were histologic type rather than tumor size correlated. Finally, papillary microcarcinoma may develop in TSHoma patients due to long term TSH overstimulation.

PE16

THYROID PAPILLARY CARCINOMA ARISING IN ECTOPIC THYROID TISSUE WITHIN RIGHT SUPRACLAVICULAR NECK AREA

1YIN-HUEI CHEN, 1CHING-CHUNG CHANG, 1GIA-YIN KUO, 2CHUN-CHIEH YEH, 4CHEN-YUAN LIN, 3YEN-NIEN LIN, 1CHWEN-TZUEI CHANG

1Division of Endocrinology and Metabolism, Department of Internal Medicine, China Medical University Hospital; 2Department of Surgery, China Medical University Hospital, Taichung, Taiwan; 3Division of Cardiovascular Medicine, Department of Medicine, China Medical University Hospital; 4Division of Hematology and Oncology, Department of Medicine, China Medical University Hospital, Taiwan

Background: More than 99% of thyroid cancers arise eutopically within the thyroid gland. Thyroid ectopy has a reported prevalence of approximately 1 in 10,000. Lingual, sublingual, thyroglossal, laryngotracheal, and lateral cervical are the most frequent origins of ectopic thyroid tissue. Ectopic thyroid tissue can be subject to the same pathological processes as normal eutopic thyroid tissue such as inflammation, hyperplasia, and tumorigenesis. Notably, regarding malignant lesions of the ectopic thyroid, it may be extremely difficult to distinguish primary neoplastic involvement of ectopic thyroid tissue from a cervical lymph node metastasis.

Case presentation: A 32-years-old female was incidentally found to have right neck mass during health examination. She was referred to endocrinologist and physical examination was remarkable for one elastic mass, measured 3.0cm in right neck. Thyroid sonography disclosed a aspiration cytology showed no evidence of malignancy, she was referred to surgeon for malignant like sonographic appearance. The patient underwent a right total thyroidectomy and left subtotal thyroidectomy. Histology showed a multifocal papillary carcinoma with follicular variant. She then received further left total thyroidectomy and a subsequent I131 ablation.

Conclusion: An ectopic thyroid cancer should be considered in the differential diagnosis of a pathological mass in the neck. Treatment of ectopic cervical thyroid cancer is based predominantly on the surgical excision of the malignant lesion. Management strategies, including performance of total thyroidectomy, neck dissection, and treatment with radioiodine, should be based on individualized risk

PE17

UTILIZATION OF RADIOFREQUENCY ABLATION FOR TREATMENT OF LOCALLY RECURRENT PAPILLARY THYROID CANCER WITH METASTATIC LYMPH NODE- A CASE SERIES AND LITERATURE REVIEW

1WEN-CHIEH CHEN, 2WEI-CHE LIN, 1JUNG-FU CHEN, 1CHEN-KAI CHOU

1Division of Metabolism, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan; 2Department of Diagnostic Radiology, Kaohsiung Chang Gung Memorial Hospital, and Chang Gung University College of Medicine, Kaohsiung, Taiwan.

Background: Surgery is the standard management for recurrence of differentiated thyroid and for those with high surgical risk or refuse surgery. Considering the above condition, radiofrequency ablation (RFA) has been suggested to be a safe and well-tolerated alternative by American Thyroid Association (ATA). In the mean time, the efficacy of RFA for lymph node metastasis refractory to radioiodine therapy is not fully understood.

Methods: We present three cases of treatment of recurrent tumor or metastatic lymph node treated with the use of RFA session(s) in patient with previous history of total thyroidectomy and neck lymph node dissection for papillary thyroid carcinoma.

Results: We present case 1 as a 63-year-old male found a mediastinal mass with size of dissection, with negative surgical margins (R0). The pathological result yielded papillary carcinoma (PTC), pT4aN1bM0 (AJCC, 7th edition), stage IVa. He received radioiodine therapy twice with biochemical incomplete (accumulated dose 240 mCi, stimulated thyroglobulin was 18.9 and 10.2 ng/ ml, separately). Two years later a RAI-refractory lesion in thyroid bed was noted. The patient received after the second RFA session). Shrinkage of tumor size (from 0.7 to 0.6cm) on US was also noted.

Case 2 is a 42-year-old female with PTC (AJCC 7th pT4aN1b), post total thyroidectomy with lymph node dissection. For metastatic papillary carcinoma, she took radioiodine therapy (twice, accumulated 250mCi); elevated Tg level remained and refractory to RAI was suspected by PET. Therefore, she received RFA session twice (Tg 79.2 ng/ml before RFA). Shrinkage of tumor size over left thyroid bed, isthmus, left neck level III and right paratracheal space was noted with improved Tg level to 40.7 ng/ml at 1 month after RFA. For one newly-found right supraclavicular metastatic lesion, repeated RFA or surgical intervention may be needed.

Case 3 is a 56-year-old male with PTC post total thyroidectomy and radioiodine therapy at other hospital. During regular follow-up, one new nodule with size of 9.1x3.3x7.6 mm in left paratracheal region with suspicion of malignancy on biopsy was noted. He then received RFA session in Nov, 2017

Conclusions: The RFA session can lead to biochemical (Case 1, 2) or structural (Case 1, 3) tumor location, surgical intervention is still the standard management. In conclusion, radiofrequency ablation is safe and well-tolerated, with a low incidence of complication, and is an appropriate method for those not suitable or refuse of operation.

PE18

ECTOPIC PARATHYROID ADENOMA: A CASE REPORT

1CHIEH CHANG, 1NING-CHUN WENG, 2YI-SUN YANG, 2EDY KORNELIUS, 3WEISHIN CHEN, 2SHIH-CHAN LO, 2CHIEN-NING HUANG

1 Medical School, Chung-Shan Medical Unversity, Taiwan 2Division of Endocrinology and Metabolism, Department of Internal Medicine, Chung-Shan Medical University Hospital, Taiwan 3 Department of Surgery, Taichung General Veterans Hospital, Taiwan

Ectopic parathyroid glands result from aberrant migration during early stages of development and lack of successful identification may lead to lack of success in parathyroid surgery. They constitute a common etiology of persistent or recurrent hyperparathyroidism, when they are missed at initial diagnosis. We describe a case of a 35-year-old male presenting with ankylosing spondylitis, incidentally found to have hypercalcemia (calcium level 13.8 mg/dl) during routine follow up of ankylosing spondylitis. Subsequent evaluation revealed elevated parathyroid hormone level (1675 pg/ ml). Suspecting hyperparathyroidism, Technetium-99 (Tc-99m) sestamibi scan was done, revealing an abnormal persistent hot spot at right thoracic inlet: high right paratracheal region behind right sternoclavicular joint, suspected an ectopic parathyroid adenoma. CT scan of neck revealed a 3.1 cm nodular lesion over right upper paratrachea region. The mass was surgically excised and it was diagnosed as a parathyroid adenoma. Serum calcium and parathyroid hormone level decreased Ectopic inferior parathyroid glands are most frequently found in the anterior mediastinum, while the most common position for ectopic superior parathyroids is the tracheoesophageal groove and After successful localization, assisted by rapid parathyroid hormone measurement postoperatively,

PE19

OVERVIEW OF STIFF PERSON SYNDROME AND VARIANTS PROVIDE AN INSIGHT INTO THE ANTI-GAD ANTIBODY TITERS AND OTHER AUTOIMMUNITY DISEASES

1YI-YIN LEE, 1YI-WEN CHEN, 1CHIH-CHING WANG, 1SZU-TAH CHEN

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Chang Gung Memorial Hospital, Chang Gung University, Taiwan, R.O.C

Background: Stiff person syndrome (SPS) is a rare neurological disorder identified as an autoimmune or paraneoplastic syndrome with frequent positive serum anti-glutamic acid decarboxylase (GAD) antibodies (anti-GAD Ab). GAD is the enzyme that catalyzes the production of that anti-GAD Ab are also detected in up to 80% of early stage type 1 diabetes mellitus (T1DM), it is of autoimmune endocrine disorders with anti-GAD Ab in SPS subjects.

Methods: This retrospective study was approved by the institutional review board of Chang Gung memorial hospital (CGMH). Patients diagnosed as SPS in CGMH since January 2001 to June 2018 were enrolled. The clinical presentation, coexisting diseases associated with anti-GAD Ab (especially T1DM and autoimmune thyroid disease, AIT), and/or other immunological (anti-nuclear antibodies, ANA) abnormalities were verified. Co-existing malignancies, major complications and medical treatment were also recorded. Anti-GAD Ab were measured by an enzyme-linked immunosorbent assay (ELISA) kit with the cut-off value of <5 U/ml.

Results: A total of 14 SPS patients (5 female and 9 male) were enrolled. The median age at symptom onset was 42.3 (19.1-84.0) years with median follow-up duration of 8.0 years. 11 patients had stiffness and painful spasms in axial muscles with progress to proximal leg muscles, 3 patients involved only bilateral lower extremities. Some patients showed symmetric spine deformity, hyperlordotic spine with limited flexibility. Nine of these patients received anti-GAD Ab tests, and among the 3 seropositive patients, 2 were concomitant with T1DM, and the other one has co-existing AIT. Another T1DM patient was diagnosed and followed-up at other hospital without available antiGAD Ab data. In the other patients with seronegative anti-GAD Ab, 1 Sjögren syndrome (ANA positive), 1 type 2 DM (onset at age 39 with insulin injection since age 49 but with normal c-peptide level) and 2 associated neoplasms (1 lung cancer and 1 thymoma) were documented.

Conclusions: Despite that T1DM and SPS are both autoimmune diseases characterized with positive anti-GAD Ab, the concurrence of SPS, T1DM and/or AIT remains sparse. The titers of anti-GAD Ab in those with SPS are much higher than those in T1 DM alone, often with a 100- to 500-fold difference. By recognizing the association of SPS with autoimmune diseases and/or malignancy, physicians may predict the development of T1DM and prevent its co-morbidities in SPS patients, and vice versa.

PE20

TRANSFORMATION OF NONFUNCTIONAL PANCREATIC NEUROENDOCRINE TUMOR INTO FUNCTIONAL PANCREATIC NEUROENDOCRINE TUMOR (MALIGNANT INSULINOMA): THREE CASES FROM A SINGLE TERTIARY CENTER

1,2SHEAU-FANG PAN, 1,3LIANG-YU LIN

1Division of Endocrinology and Metabolism, Department of Medicine, Taipei Veterans General Hospital, Taipei, Taiwan; 2Department of Internal Medicine, City Hospital of Yang-Ming Campus, Taipei, Taiwan; 3 Faculty of Medicine, School of Medicine, National Yang-Ming University, Taipei, Taiwan.

This report presented three patients of nonfunctional pancreatic neuroendocrine tumor (PNET), their initial clinical presentation with mass effects and their pathologic findings. They denied any symptom or sign of hypoglycemia. Most importantly, all three patients developed hyperinsulinemic hypoglycemia which lead to the diagnosis of functional pancreatic neuroendocrine tumor after different treatments. while other endocrine profiles were within normal limits. Their CT scan of abdomen all reported pancreatic tail tumor with liver metastasis, with one patient also having spleen invasion and another patient having lymphadenopathy. They received different systemic treatments for PNETs. But they all had tumor recurrence and developed severe symptomatic hypoglycemia later (form 1 year 3 months to 4 years 9 months) with lab data proven hyperinsulinemic hypoglycemia a. These data suggest that these three patient with initially diagnosis of nonfunctional PNET had later transformed into functional pancreatic neuroendocrine tumors. This might lead further evaluations for manipulating beta cell function.

PE21

PROPYLTHIOURACIL FROM RECTAL ADMINISTRATION IN HYPERTHYROID PATIENT

1SHENG-PANG HSU, 1CHING-CHU CHEN

1Division of Endocrinology and Metabolism, Department of Internal Medicine, China Medical University Hospital

Case Report: This 61-year-old retired male patient was admitted to MICU due to massive oral bleeding with conscious change on 2018/09/30. He was a case of left buccal SCC s/p operation (composite resection and marginal mandibulectomy and inferior maxillectomy and left SND + free treated by palliative chemotherapy and target (cetuximab)therapy recently.

In order to control the massive oral bleeding, transarterial embolization (TAE) which showed acute blow-out of the right proximal external carotid artery post selective transarterial embolization. After TAE .The bleeding was controlled and Hemoglobin showed stable(6.9 g/dL-- > 10.7 g/dL).

Amiodarone IV infusion was given for Atrial fibrillation with RVR (HR:140-160) in the beginning of MICU hospitalization . Thyroid function showed thyrotoxicosis related to Graves’ disease ( TSH < 0.005 uIU/mL, free T4:3.45 ng/dL, Anti-TPO:319 IU/mL, Anti-thyroglobulin antibody: 28.7 IU/mL, TSH-recepter antibody: 32.8%). However, patient has no NG tube due to high risk of tumor bleeding. The General Surgeon planned to perform gastrostomy after relative stable hemodynamic 2018.10/01-10/08(total 16 times of enema plus IV esmolol). Gastrostomy was performed on 10/08 uIU/mL, free T4: 2.14 ng/dL and PTU shifted to PTU to 150mg BID PO. Thyroid function on 10/31 showed TSH < 0.005 uIU/mL, free T4: 1.11 ng/dL with relative stable heart rate around 80-100 times/ min.

Discussion: insertion, how could anti-thyroid drugs be given become a problem. However, PTU and methimazole enema are effective and safe treatment after review the literature. The detail results of literature review would show on poster.

Conclusion: Propylthiouracil enema is a effective and safe treatment while patient can’t prescribe anti-thyroid drug from oral or NG route.

PE22

CONCOMITANT PRESENTATION OF DIABETIC KETOACIDOSIS AND THYROTOXICOSIS IN A PREGNANT WOMAN- A CASE REPORT

1YING-LEIN CHENG,

1YU-MEI CHIEN, 1.2TING-WEI LEE, 1HAN-WEN LAO, 1YU-CHUN HSUEH, 1.3TING-I LEE

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Taipei Medical University-Wan Fang Municipal Hospital, Taipei, Taiwan; 2Graduate Institute of Clinical Medicine, School of Medicine, College of Medicine, Taipei Medical University, Taipei, Taiwan; 3Department of General Medicine, School of Medicine, College of Medicine, Taipei Medical University, Taipei, Taiwan

Diabetic ketoacidosis (DKA) is an acute complication that may have devastating consequences if not managed promptly and effectively. DKA is mostly seen in type 1 diabetes mellitus (DM) patient. Although simultaneous presentation of thyrotoxicosis and DKA have been reported, it is a clinically that remains a diagnostic and management challenge in clinical practice. Herein, we described a rare of concurrent presentation of a newly diagnosed DM complicated with DKA and thyrotoxicosis in a pregnant women.

A 40-year-old women with intrauterine pregnancy 33 weeks presented to our Emergency room due to back pain, diarrhea, nausea and vomiting for few hours. Seven days prior to visit, she had common cold and headache with medication from local clinic. Her Gestational history was G4P1SA1AA1 with normal prenatal physical examination. 100g oral glucose load at 28th weeks of gestation exclude diagnosis of gestational DM. She denied past medical history. She had cesarean section five years ago. She denied of any heredofamilial disease. On physical examination, height was 166 cm and body weight 63 kg with body mass index 22.8 kg/m2 (previous BW 52kg). She is afebrile with blood pressure 160/79 mm Hg, heart rate 140 beats/min, respiratory rate 27 breaths/min and 97% O2 saturation at room air. Her skin was dry, with vitiligo. Thyroid gland was diffuse grade 2 Unfortunately, absent of fetal heart beats was found at the Emergency room, and she underwent emergent cesarean section, and delivered a stillborn baby girl. Laboratory results revealed random blood glucose of 418mg/dL, high anion gap metabolic acidosis (pH of 7.24, HCO37.6 mmol/l, anion gap of 20.4 mmol), and ketone body 5mmol/l. Her renal, liver function, and electrolytes were normal. The patient was treated as DKA. Additional investigation showed HbA1c 6.3%, fasting C-peptide was 0.4 ng/ml, tyrosine phosphatase antibodies was negative but positive glutamic acid decarboxylase antibodies. Glucagon stimulation test showed blood glucose and C-peptide at 0’ and 6’ were 210 mg/ dl and 234mg/dl, and 0.26 ng/ml and 0.71 ng/ml, respectively. Type 1 DM was highly suspected and 2 goiter, thyroid was further evaluated. Results showed serum FT4 2.43 ng/dl, TSH 0.04 uIU/ml, Her

anti-thyroglobulin antibodies was negative, but positive for anti-thyroid peroxidase antibodies and TSH receptor antibody. Follow-up of thyroid functions showed T3 449.14ng/dl, FT4 4.33ng/dl, and she was treated as Graves’ disease in thyrotoxic state.

PE23

A NEW BIOCHEMICAL SCREENING TEST FOR THE DEFINITIVE DIAGNOSIS OF ENDOGENOUS CUSHING’S SYNDROME: MORNING LOW-DOSE DEXAMETHASONE SUPPRESSION TEST

SHIH-CHEN TUNG, PEI-WEN WANG

Kaohsiung Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Kaohsiung, Taiwan

Introduction: Endogenous Cushing’s syndrome (CS) is a rare severe condition resulting from a chronic excessive endogenous glucocorticoid secretion that causes systemic morbidity and a higher mortality risk. The definitive diagnosis of endogenous CS is a challenge in clinical endocrinology. (UFC), plasma cortisol circadian rhythm assessment, low-dose dexamethasone suppression test (LDDST), and mid-night plasma cortisol or late-night salivary cortisol levels.

Now, we present a new biochemical screening test, e.g. morning low-dose dexamethasone

Materials and methods: From June 2001 to January 2017, endogenous CS was diagnosed in 24 subjects who morning LDDST (1 mg dexamethasone administered orally at 07:00 h and plasma cortisol withdrawn at 16:00 h) was performed. Other traditional screening tests for CS were also performed including baseline morning (08:00 h to 09:00 h) and afternoon (16:00 h to 22:00 h) plasma cortisol levels, baseline 24-h UFC, and overnight and 2-day LDDST. 7.5 mg/dl and > 50% of baseline morning plasma cortisol level. Non-suppression of morning LDDST is defined as afternoon plasma cortisol level at 16:00 h > 5 mg/dl. Non-suppression of overnight LDDST (administration of 1 mg dexamethasone orally at 23:00 h) is defined as morning 08:00 h plasma cortisol level > 5 mg/dl. Non-suppression of 2-day LDDST (0.5 mg dexamethasone orally h plasma cortisol level at the third day > 5 mg/dl. Plasma cortisol and UFC were measured by radioimmunoassay (RIA) kits. Plasma ACTH levels were measured by RIA or immunoradiometric assay. Results: The mean age of these 24 patients at the time of surgery was 43.4 ± 11.9 years old. Sex distribution is 19 females and 5 males, respectively. The distribution of the causes of CS is 17 patients with adrenal CS (12 patients with adrenocortical adenoma, one with adrenocortical carcinoma, two with primary pigmented nodular adrenocortical disease, and two with ACTH-independent bilateral disease and two with ectopic ACTH syndrome).

The sensitivity of biochemical screening tests for CS with morning LDDST is 91.7% (22/24),

overnight LDDST 95.5% (21/22), loss of diurnal rhythm 95.8% (23/24), baseline 24-h UFC 100% (19/19), and plasma cortisol at 08:00 h after 2-day LDDST 91.7% (11/12).

Conclusions: 1. The sensitivity of the screening test for endogenous CS with morning LDDST is 91.7%, not higher or lower rate as compared with other traditional reported screening tests of CS 2. It is more convenient for patients to go to hospital afternoon for withdrawing blood with morning LDDST.

PE24

TYOSINE KINASE INHIBITORS AND MTOR INHIBITORS RELATED ENDOCRINOPATHY: A CASE REPORT

1JIA-YIN GUO, 1TZU-YUAN WANG, 1RONG-HSHING CHEN, 1CHWEN-TZUEI CHANG, 1SHENG-PANG HSU, 1CHING-CHU CHEN, 1CHING-CHU CHEN

1Division of Endocrinology and Metabolism, Department of Medicine, China Medical University Hospital, Taichung, Taiwan, R.O.C

Introduction: There was an enhancing improvement for cancer treatment in the past decades. Among medicines for target therapy, tyrosine kinase inhibitors interfere with molecules essential for tumor growth and mTOR inhibition results in growth suppression. Here, we reported a case using a tyrosine kinase inhibitor and a mTOR inhibitor for treatment of renal cell carcinoma leading to hypothyroidism and diabetes mellitus.

Case report: A 66-year-old male with a history of right renal cell carcinoma (RCC) was first seen at our endocrine out-patient clinic due to referral for management of hypothyroidism. He was diagnosed to have right RCC 9 years ago, received a right radical nephrectomy. Three years ago, the RCC relapsed with lung metastasis. He received palliative radiotherapy and target therapy with Sunitinib, a tyosine kinase inhibitor, for nearly two years, then switching to axitinib, another tyosine kinase inhibitor, due to bone metastasis one year ago. Ten months ago, thyroid function showed free T4 0.45 ng/dL and TSH 93.52 uIU/Ml, respectively. Both ATA and anti-TPO antibody were negative. Thyroid ultrasonography showed atrophic thyroiditis. Four months ago, he was found to have prostate malignant epithelioid tumor. Axitinib was replaced with everolimus, a mTOR inhibitor, for prostate cancer therapy. Three months later, hyperglycemia was noticed. HbA1c level was 6.8% and 10% in 2 consecutive three- month interval check-up. He was treated with insulin after metformin monotherapy failure.

Discussion: Tyrosine kinase inhibitors block both vascular endothelial and platelet-derived growth factor receptor to inhibit angiogenesis, which may cause hypothyroidism. The possible mechanism of mTOR inhibitors associated diabetes may be secondary to a reduction of the insulin signaling pathway leading to insulin resistance and a reduction of insulin secretion via a direct effect on the pancreatic beta cells.

Conclusion: It is needed to monitor endocrine functions during target therapy with tyrosine kinase inhibitors and mTOR inhibitors.

PE25

THYROID INFLAMMATION AND INTRA-NODULAR HEMORRHAGE AFTER FINE-NEEDLE ASPIRATION FOR THYROID CYST: A CASE REPORT

1CHI-LUNG TSENG, 2CHUN-JUI HUANG

1Division of Gastroenterology, Department of Internal Medicine, Taoyuan General Hospital, Ministry of Health and Welfare, Taoyuan, Taiwan; 2 Division of Endocrinology and Metabolism, Department of Medicine, Taipei Veterans General Hospital, Taipei, Taiwan

Background: simple procedure for managing thyroid cysts. Post-aspiration complications are rare, and pain and minor hematomas are the most common. Large intra-nodular hemorrhage is infrequently seen, and

Clinical case: A 58-year-old Taiwanese male presented to the emergency department with fever a long history of multi-nodular goiter, diabetes mellitus, hypertension, and hepatitis B carrier. Rapid enlargement of thyroid nodule was noted in September, 2017, with largest one measuring 9.0 cm over right thyroid. Since then, he started to receive fine needle aspiration fluid drainage 21-50 c.c. each time every two to three months for his thyroid cyst. Surgical intervention was suggested but the patient refused. In August, 2018, another 50 c.c. of brownish cystic fluid was drained for a 5.6 cm thyroid cyst. Unfortunately, fever and enlargement of right neck mass developed three days afterwards. His vital signs were temperature 38.5°C, heart rate 102 beats per minute, respiratory rate 20/min, blood pressure 146/88 mmHg. Physical examination revealed right side stony-hard thyroid with tenderness. Hemogram and thyroid function were normal but markedly elevated C-reactive protein level to 19.3 mg/dL (normal: 0.0-0.5) was found. There was no evidence of infection on urine routine, CXR, abdominal sonography, and blood cultures. Neck computed tomography (CT) scan disclosed multi-nodular goiter with intra-nodular hemorrhages up to 5 cm in right thyroid. Findings on thyroid sonography were compatible. Intravenous antibiotic with Piperacilin/tazobatam was administered for suspicious acute suppurative thyroiditis and later shifted to cefepime plus metronidazole. Fine needle aspiration drainage was performed over right thyroid, and the bacterial culture, tuberculosis smear and culture of the 25 c.c. bloody aspirate were negative. Fever and neck pain persisted despite antibiotics also elevated level of erythrocyte sedimentation rate (ESR) to 83 mm/hr (normal: 0-20). Therefore, we treated him as subacute thyroiditis with prednisolone 20 mg B.I.D. Fever, neck pain and swelling improved and prednisolone dosage was tapered. Twenty days later, ESR normalized to 18 mm/hr.

Clinical lesion: Although fine needle aspiration of the thyroid is a relatively safe procedure, with frequent aspirates of large amounts.

PE26

A CASE OF ONCOCYTIC ADRENOCORTICAL CARCINOMA WITH FUNCTIONING CLINICAL PRESENTATION

1SHIH-PEI CHEN, 1YI-HONG ZENG, 1CHUN-CHUAN LEE

1Division of Endocrinology and Metabolism, Department of Internal Medicine, MacKay Memorial Hospital, Taipei, Taiwan

Oncocytic adrenocortical carcinoma is a rare histopathological variant of adrenocortical carcinoma with very few instances reported in the literature to date. We report a case of oncocytic adrenocortical carcinoma with functioning clinical presentation. 36-year-old female complained of body weight gain 10 kg within 1 month, leg edema, acne over neck and forehead. In addition, insomnia, dizziness, headache, and hypertension (systolic blood pressure 160mmHg at home) were noted. Due to abdominal pain, she went to our outpatient department for help. Lab data showed WBC:6.9 10^3/uL, Hemoglobin:14.5 g/dL, Platlet221 10^3/uL , blood sugar 100mg/dL, GPT 20 IU/L,creatinine 0.8 mg/dL , Na/K 144/3.8mEq/L, Ca 9.3 mg/dL , Aldosterone 5.74ng/dL, Plasma dL. Computerized tomography scan revealed a 6.7cm heterogeneously right adrenal mass mass with possible thrombus within IVC at the level of right adrenal tumor. Positron Emission Tomography showed a large mass of markedly, heterogeneously increased tracer uptake (SUVmax: 15.3) in right adrenal region with slightly decreased delayed phase radioactivity (SUVmax: 14.1). She underwent operation of excision right retroperitoneal malignant adrenal tumor with partial right nephrectomy and excision part of IVC. Pathology was proven right adrenal cortical carcinoma, oncocytic variant. Then, she received adjuvant radiotherapy and Mitotan.

PE27

A CASE OF PRIMARY HYPERPARATHYROIDISM WITH VITAMIN D DEFICIENCY

HUI-I YU, FANG-PING KUNG, TSAI-SUNG TAI

Ditmanson Medical Foundation Chia-Yi Christian Hospital

A 65-year-old male, presented with histories of hypertension, renal stone, chronic constipation, insomnia. He had ever admitted due to acute pancreatitis and high calcium level 12 mg/DL and I-PTH being 961 pg/ml was shown. Then he was referred to our hospital. Laboratory investigations revealed Alb: 3.3, Total calcium: 14.4, Ion.Ca:2.03, P:1.0, I-PTH:1149.1, Na:131mmol/L, K:3.31mmol/l, mg: 1.87. Low plasma 25(OH)D3 levels, 25(OH)D3 being 9 ng/ml (normal values <30 ng/ml) was observed. Thyroid ultrasound found cystic goiter and parathyroid adenoma, right 26.3x20.3x18.3mm. Tc-99m T1-201 Subtraction scan showed parathyroid adenoma in the inferior pole of right thyroid lobe. Bone mineral density study was measured in the neck of the left femur and L-spine received right parathyroidectomy. Post-Operation his calcium and phosphate level returned to normal, Calcium level being 9.2 mg/dl, Phosphate level being 3.8 mg/dl. His I-PTH level dropped to 23.7 pg/ ml. He received temporarily calcium supplement and maintain Vitamin D supplementation without hungry bone syndrome.

Conclusions: primary hyperparathyroidism (PHPT) than in general population. It is often associated with more severe PHPT with higher PTH level or bigger size of adenoma. Cautious vitamin D supplementation before operation does not aggravate hypercalcemia and might reduce the risk of hungry bone syndrome following surgery. Thus, Vitamin D status assessment in patients with PHPT is important.

PE28

PULMONARY EMBOLISM AS THE INITIAL PRESENTATION OF ADRENAL CUSHING SYNDROME

1YU-CHIEH CHANG, 1CHUN-CHUAN LEE, 1YI-HONG ZENG

1Division of Endocrinology and Metabolism, Department of Internal Medicine, MacKay Memorial Hospital, Taipei, Taiwan

Background: Cushing syndrome is associated with a hypercoagulable state results in a 10-fold increase in the incidence of venous thromboembolism(VTE) compared with the general population. gene transcription. The hypothesis is supported by the increase in coagulation factors with subsequent shortening of aPTT and normalization after treatment. Pulmonary embolism and deep venous thrombosis are the two most important manifestations of venous thromboembolism (VTE). We hereby report a case with pulmonary embolism as the initial presentation of Cushing syndrome.

Case Report: A 63-year-old female with type 2 diabetes and hypertension presented to our emergent department with chest tightness and shortness of breath. Her vital signs: body temperature 37.3 , pulse 100/min, respiratory rate18/min, blood pressure: 149/83 mmHg. The patient has the appearance of central obesity with several ecchymosis over her limbs and trunk. Laboratory investigation showed white blood cell count 8700/uL, segment 59%, PT 11 sec, aPTT 25.5 sec, creatinine 0.9 mg/dL, sodium 141 mEq/L , potassium 2.5 mEq/L, GPT 66 IU/L , troponin-I 0.57 ng/ mL, CKMB 6.3 ng/mL, BNP 297 pg/mL and D-dimer 5671 ng/mL. Arterial blood gas showed pH 7.506, pCO2 27 mmHg, pO2 72 mmHg, HCO3 22 mmol/L, B.E. 0.5 mmol/L, SaO2 96%. EKG showed sinus tachycardia. Chest x ray showed cardiomegaly with engorged pulmonary trunk. and inferior branches of bilateral pulmonary arteries, indicating pulmonary embolism. Furthermore, a 3cm heterogenous mass of left adrenal gland was noted. Venous ultrasound showed thrombosis over the right popliteal vein causing partial occlusion. The patient was given anticoagulant and was closely monitored in our intensive care unit (ICU). 88.3 ug/day, urinary vanillylmandelic acid(VMA) test 5.8 mg/day. Cortisol of overnight dexamethasone suppression test, low-dose dexamethaone suppression test, high-dose dexamethasone suppression syndrome was confirmed and she received laparoscopic left adrenalectomy with pathology reported

Conclusion: The etiology of Cushing syndrome includes corticotrope adenomas in the pituitary (Cushing’s disease), ectopic corticotropin (ACTH) production by tumors (eg, small cell carcinoma), or adrenal adenomas and carcinoma. Pulmonary embolism is a peculiar initial presentation of Cushing syndrome. It is suggested that Cushing syndrome should be considered in a patient with hallmark manifestations and pulmonary embolism.

PE29

MANAGEMENT OF COMPLICATED THYROTOXICOSIS WITH EXTRACORPOREAL MEMBRANE OXYGENATION

1SZU-HAN LIN, 1SHANG-REN HSU, 1YU-FANG CHENG, 1PO-CHUNG CHENG, 1DONG-HWA TSAI

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Changhua Christian Hospital, Changhua City, Changhua County, Taiwan

Uncontrolled thyrotoxicosis may have catastrophic consequences. During the year 2018, three hyperthyroid patients were admitted to our hospital for serious complications that required life support with extracorporeal membrane oxygenation (ECMO).

Case 1 is a 35-year-old man who had bilateral proximal lower limb weakness in the morning and was discovered to have hypokalemia. While at the emergency department, refractory ventricular tachycardia developed, leading to cardiac arrest and initiation of ECMO. The patient was found to be complicated with rebound hyperkalemia following potassium replenishment. After appropriate treatment, the patient stabilized and was freed from ECMO.

Case 2 is a 55-year-old woman with Graves’ disease managed with subtotal thyroidectomy more than 20 years ago. She presented with worsening exertional dyspnea, leg edema, and palpitations of for cardiorespiratory failure which required endotracheal intubation. After admission she had cardiac arrest due to ventricular tachycardia and was supported with ECMO. Hyperthyroidism was diagnosed as well as severe mitral regurgitation and poor ventricular function. After anti-thyroid drug treatment and heart valve replacement surgery, the patient improved and was discharged in stable condition.

Case 3 is a 28-year-old man with irregularly treated Graves’ disease who presented with chest tightness since the day before. Goiter and elevated thyroid hormones were found. Initial electrocardiogram revealed intermittent short run ventricular tachycardia. Coronary angiography was performed, which showed left anterior descending artery myocardial bridging. The patient was discharged following treatment with anti-thyroid drug and Lugol’s solution. However, he was readmitted 6 days later for thyrotoxic crisis and refractory ventricular tachycardia requiring initiation of ECMO. Escape from the inhibitory effect of iodine load on thyroid function was considered a plausible cause. After appropriate anti-thyroid treatment, the patient was successfully freed from ECMO.

These cases demonstrate the usefulness of ECMO as a lifesaving measure for patients with other aggravating conditions.

PE30

LITHIUM-INDUCED HYPERTHYROIDISM: A CASE REPORT

1CHIN-CHOU YANG, 2PENG-NIEN WANG

1Division of General Medicine, Department of General Psychiatry, Tsaotun Psychiatric Center, Ministry of Health and Welfare, Nantou, Taiwan; 2Department of General Psychiatry, Tsaotun Psychiatric Center, Ministry of Health and Welfare, Nantou, Taiwan

Background: Lithium is widely used as a mood stabilizer in managing mental illness, mainly mania and bipolar disorder. In this article, we report a 38 years old woman admitted for schizophrenia. Lithium-induced hyperthyroidism was found during admission.

Methods: A 38 years old woman without systemic disease was admitted to our psychiatric ward under the diagnosis of schizophrenia. The routine laboratory study revealed normal thyroid , Clonazepam, Biperiden, and Lithium. These medications remained the same in recent one year. In August 2018, abnormal thyroid function was found. (TSH < 0 uIU/ml, FT4 2.35 ng/dl). Thyroid antibodies were within normal range (TSHR-Ab negative, anti-TPO Ab: 31 IU/mL). The patient denied any discomfort. So we kept closely observation and followed thyroid function 1 month later. The data revealed: TSH < 0 uIU/ml, FT4 2.59 ng/dl. The tentative diagnosis was Lithium-induced hyperthyroidism. Methimazole was prescribed for hyperthyroidism treatment.

Results: The thyroid axis is prone to interactions with many drugs, and hypothyroidism is the most common condition of drug-induced thyroid dysfunction (1). Lithium is wildly used as treatment for schizophrenia and is often associated with a decline in renal function, hypothyroidism, and hypercalcemia (2). Thyroid disorders such as goiters and hypothyroidism are common complications of the use of lithium. Although the occurrence of lithium-induced hyperthyroidism is rare, it has still been reported (3). The mechanism of lithium-induced hyperthyroidism is not well understood. However, this condition can generally be controlled medically and there is no need to discontinue

Conclusions: We report a case of lithium-associated hyperthyroidism. The condition was resolved by Methimazole. It is strongly recommended to evaluate thyroid function before starting to treat psychic patients with Lithium. If hyperthyroidism develops, treatment should be carried out according to guidelines, and it does not always require withdrawal of lithium.

References:

1. RIZZO, Leonardo FL; MANA, Daniela L.; SERRA, Héctor A. Drug-induced hypothyroidism.

Medicina (Buenos Aires), 2017, 77.5: 394-404. 2. SHINE, Brian, et al. Long-term effects of lithium on renal, thyroid, and parathyroid function: a retrospective analysis of laboratory data. The Lancet, 2015, 386.9992: 461-468.

3. CHALASANI, Santhi; BENSON, Katherine A. Lithium-induced thyrotoxicosis in a patient with treatment-resistant bipolar type I affective disorder. Med J Aust, 2014, 201.9: 541-542.

PE31

PAPILLARY THYROID CARCINOMA COMBINED WITH SKIN DISTANT METASTASIS

1CHING-LING TU,

1HSING-YI HUANG, 1HE-JIUN JIANG, 1WEI-WEN HUNG, 1PI-JUNG HSIAO, 1SHYI-JANG SHIN, 1MEI-YUEH LEE

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Kaohsiung Medical University Hospital, Taiwan, R.O.C

Papillary thyroid carcinoma has three types of tumor recurrences including postoperative nodal metastases, local recurrence, and postoperative distant metastases. Papillary thyroid carcinoma usually has outside regional lymph nodes metastasis and lung distant metastasis. Fewer than 1% of all patients with papillary carcinoma, had cutaneous metastases. We report a case of papillary thyroid carcinoma with lymph-vascular invasion and minimal extrathyroid extension, post total thyroidectomy and I-131 therapy initially. However, post-operation thyroglobulin level kept elevating. Thyroid echo found residual thyroid tissue and neck CT found residual thyroid tissue and multiple nodules in the right anterior lower neck. Radical thyroidectomy with unilateral lymph node dissection, and skin and carcinoma with lymph nodes and skin and subcutaneous metastasis.

Case presentation: This 42 years old female patient didn’t have any systemic disease, who presented with palpable anterior neck mass near right supraclavicular area for 2 months with symptoms including foreign body sensation and mild compression feeling while swallowing in 2014.The mass did not induce pain.She denied dysphagia, odynophagia, hoarseness, dyspnea, fever, sore throat, cough, or other common cold symptoms. She also denied palpitation, easily sweating, hands tremors, and body weight loss. She never smoked, and denied alcohol and betel nuts use. About her family history, her cousin had unknown thyroid disease.

Lab data showed normal thyroid function (TSH: 4.87uIU/ml(0.25-4) , Free T4: 1.03ng/dl(0.71.8)) but elevated thyroglobulin level (Thyroglobulin: 54.61ng/ml(< 50)). thyroid echo found right single hypo-echoic nodular goiter (1.86cm*1.07cm). Thyroid fine needle aspiration (FNA) was done and FNA cytology report was nondiagnostic result ((Bethesda System for Reporting Thyroid Cytopathology; diagnostic category I).)

We recommended rearrange thyroid FNA and recommended patient to avoid Iodine-rich foods. But the patient lost follow-up. She finally came back 2 years later due to referral from our GS(general surgeon) department. The patinet’s thyroid nodule enlarged to more than 4cm, so the thyroid carcinoma (PTC): a 4.5cm malignant nodule at right lobe with lymph-vascular invasion and minimal extrathyroid extension (to perithyroid soft tissue or skeletal muscle). There was negative for

malignancy in a small regional lymph node in section but the surgical margin was involved. AJCC 8th staging was T3bN0AmX, stageI, ATA High Risk.

We then arranged post-operation neck sonography but no obvious remnant found. We also prescribed thyroxine for hormone supplement and therapy. Post-operation thyroglobulin (Tg) persistently maintained more than 10ng/ml(13.3ng/ml), and we arranged high-dose radioactive iodine (RAI). Subsequent whole body scan (WBS) revealed residually biological remnants in the right tubercle and pyramid, a faint radioiodine avid lymph nodes in the left supraclavicle basin (N1b), and less image evidence of I-131 avid distant metastasis.

After completed I131 therapy, out-patient-department follow-up Tg was still elevating from 13.3ng/ml to 84.8ng/ml within 6 months. Therefore, we re-arranged neck sonography that found thyroid tissue remnants this time. Neck CT with contrast revealed bilateral residual thyroid glands, multiple nodules in right anterior lower neck and borderline enlarged right neck level IV lymph nodes. Radical thyroidectomy with unilateral lymph node dissection was smoothly done; skin and nodes and skin metastasis. The patient then accepted further I131 therapy again.

Discussion: Papillary thyroid carcinoma has three types of tumor recurrences including postoperative nodal metastases, local recurrence, and postoperative distant metastases. Papillary thyroid carcinoma usually has outside regional lymph nodes metastasis and lung distant metastasis. Fewer than 1% of all patients with papillary carcinoma, had cutaneous metastases in thyroid cancer case, palpation of the thyroid bed and lymph node areas is routinely performed. Ultrasonography is more sensitive and may detect lymph nodes as small as 2 to 3 mm in diameter. Thyroglobulin (Tg) is a glycoprotein that is produced only by normal or neoplastic thyroid follicular cells. Serum Tg should not be detectable in patients who have had total thyroid ablation. Here, we presented a case Physical examination, thyroid echo, serum thyroglobulin for follow-up are needed.

PE32

78 YEARS OLD WOMAN WITH EXTREME HYPOTHYROIDISM PRESENTING WITH MYXEDEMA COMA

1LIU FEN, 1WANG HUI

1Cardinal Tien Hospital

Background: This 78 years old woman was brought to our emergency department for decreased mental status with scanty urine output for 1 day.Abnormal thyroid thyroid function hypothyroidism was noted,after thyroid hormone replacement,her consciousness become improved.

Conclusions: Myxedema coma is a true endocrine emergency requiring early and appropriate treatment. Patients presenting to the emergency department with hypothermia are rare and often require prompt diagnosis and management.

PE33

ECTOPIC PAPILLARY CARCINOMA FOUND IN GRAVES’ DISEASE: A CASE REPORT

1JIA-YIN GUO, 1CHWEN-TZUEI CHANG, 1MAO-TSU FUH, 1RONG-HSHING CHEN, 1TZU-YUAN WANG, 1WEI-LUN HUANG, 1CHING-CHUNG CHAN, 1YIN-HUEI CHEN, 1SHENG-PANG HSU, 1CHING-CHU CHEN

1Division of Endocrinology and Metabolism, Department of Medicine, China Medical University Hospital, Taichung, Taiwan, R.O.C

Introduction: Papillary thyroid carcinoma was the most common type of thyroid cancer, with a favorable prognosis. Here, we report a rare case of papillary thyroid carcinoma arising from ectopic thyroid gland in Graves’ disease.

Case report: clinic due to hyperthyroidism with initial presentation of palpitation. Physical examination showed diffuse thyroid goiter grade II, with one elastic nodules about 2 cm on right side. Thyroid function showed free T4 3.45 ng/dL and TSH 0.018 uIU/Ml. TSH Receptor antibody was 51.9%. Thyroid ultrasonography revealed one 6.1*9.3 mm hypoechoic nodules in left side. One hypoechoic nodule 10.6*12.5 mm with calcification and irregular margin was noticed in right side. Also, another two isoechoic lesions with 15.2*12.3 mm and 16.4*34.4 mm located lateral to right thyroid area and was near to internal carotid artery was noted, suspect of ectopic thyroid tissue. Fine needle aspiration showed negative of malignancy. Due to Graves’s disease with suspect ectopic thyroid gland, operation with right thyroid lobectomy, left subtotal thyroidectomy and right neck dissection to remove ectopic thyroid gland was done three months later. Unexpectedly, Pathology showed papillary thyroid carcinoma of follicular variant of right lobe, multifocal papillary microcarcinoma, arising from ectopic thyroid, p T3 according to AJCC 7th edition . Left lobectomy for complete tumor excision was done. Two weeks later, which pathology showed papillary thyroid carcinoma with follicular variant, no lymph node metastasis found. Post-operation radioiodine I-131 with 150 mci was administered. There was no recurrence noted during follow up.

Discussion: Ectopic thyroid gland may be found along the line of the obliterated thyroglossal duct, usually from the tongue to the diaphragm, which may have associated with either hypofunction or hyperfunction. Rarely, malignant neoplastic changes can occur in ectopic thyroid tissue. Surgery is a prudent choice due to the potential of malignant evolution of ectopic thyroid tissue.

Conclusion: For ectopic thyroid tissue, in addition to evaluate of hormone-producing function, elaborate ultrasonography examination for nodular disease or malignant transformation was also

PE34

A CASE OF HUGE PAPILLARY THYROID CARCINOMA IN STRUMA OVARII BUT NEGATIVE FINDINGS IN THYROID GLAND AND REVIEW OF LITERATURE

MIN-TSUN CHIU, KAI-JEN TIEN, MEI-CHEN YEH, NAI-CHENG YEH, SHANG-GYU LEE, CHEWN-YI YANG

Division of Endocrinology and Metabolism, Department of Internal medicine, Chi Mei Medical Center, Tainan, Taiwan.

Objective: Thyroid tissue is not commonly seen in ovary. Struma ovarii is diagnosed when thyroid tissue is the predominant element (> 50%) in ovarian teratoma. About 5% of all ovarian teratomas are struma ovarii. Due to the rarity of the disease, there is no consensus on the optimal treatment of women with malignant struma ovarii. Here we present a rare case of huge struma ovarii with papillary thyroid carcinoma, and we review the management and treatment option of this rare tumor.

Methods: In this article, one case of struma ovarii was reviewd. A 47-year-old female was presented to out-patient department of gynecology due to vaginal spotting. Menopausal was found for 6 years then. Before menopausal, there was dysmenorrhea and hypermenorrhea during menstral period. Transvaginal ultrasonography was arranged and showed diffused heterogeneously echogenic mass with 8.6 cm in size found in posterior uterine wall. Lab data showed elevation of tumor markers (CA125, CA199).

Results: Total abdominal hysterectomy with bilateral salpingo- oophorectomy (TAH/BSO) was done. Pathology showed papillary thyroid carcinoma. Tumor markers fell back after surgical intervention. Thyroid echo followed at out-patient department of endocrine showed no positive of tumor or malignancy. Antithyroglobulin antibody, anti-thyroid peroxidase, T4, TSH, and thyroglobulin were within normal limit.

Conclusions: The choice of optimal treatment is still uncertain. However, for malignant struma ovarii, local resection of ovarian lesion and thyroid evaluation should be done. The need for

PE35

DE MORSIER’S SYNDROME WITH DIABETES INSIPIDUS, A CASE REPORT

1CHUN FENG WU, 1PIN FAN CHEN, 1BING RU GAO, 1WEI CHENG LIAN

1Division of Endocrinology and Metabolism, Department of Internal Medicine, Dalin Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation

Background: de Morsier’s Syndrome, also called Septo-optic dysplasia (SOD) is a rare congenital abnormality and characterized by a classic triad of optic nerve hypoplasia, midline brain complicated hypopituitarism with both anterior and posterior pituitary function defects.

Case presentation: A 29-year-old visually impaired female was referred to our outpatient department for morbid obesity evaluation. Her past medical history included optic nerve dystrophy and growth retardation, which received one-year growth hormone replacement during her early adolescence. The increase in thirst (> 10 liters of water per day) and a large amount of urine amount were also noted for years. Pituitary function showed IGF-1 at very low levels, but normal prolactin, TSH, ACTH and FSH values.

At admission, we arranged the glucagon test and the water deprivation test. The diagnosis of growth hormone deficiency and central diabetes insipidus was confirmed. Brain MRI revealed 1) right optic nerve and chiasm hypoplasia, 2) thin pituitary stalk and an intermediate signal of posterior pituitary on T1, and 3) absence of septum pellucidum. We diagnosed SOD according to the MRI finding, pituitary dysfunction, and optic nerve defect. The polyuria improves after desmopressin therapy.

Conclusion: SOD was a heterogeneous disorder with multiple system involvement. According stimulating hormone, adrenocorticotropin, and gonadotropin. Deficiency of antidiuretic hormone was relatively rare in SOD. For young patients with a history of optic nerve dysfunction and growth of life.