bluuebunny/crossref_metadata_2025_split huggingface dataset:
P-35 Luteinized Thecoma in a postmenopausal woman with Hirsutism
Authors: Imre Eren 
Published: January 2024

Abstract

Introduction
We present a 59-year-old postmenopausal woman with a history of obesity and hypertension who sought medical attention due to severe hirsutism. She had been taking various medications, including levotiroksin for thyroid hormone replacement, asetyl salicylic acid as an antiplatelet agent, Betahistin for vertigo, and amlodipine 10 mg for hypertension.

Clinical Case
The patient had been experiencing severe hirsutism for a year and her complaining had been deteriorating. Her past medical history was significant for obesity and hypertension. She had undergone menopause ten years ago. Her hirsutism, and obesity prompted further investigation into her hormonal profile and pelvic health.
On physical examination, the patient's height was 153 cm, and her weight was 109.7 kg, with a BMI of 46.6. Notably, she exhibited severe hirsutism, with Feriman Gallway score of 15. The patient's laboratory results revealed TSH level was elevated at 4.98 mIU/L, indicating subclinical hypothyroidism. Further hormonal evaluation showed increased FSH levels (40 IU/L) and LH levels (12 IU/L), consistent with diminished ovarian function. DHEAS (dehydroepiandrosterone sulfate) level was 121 µg/dL, PRL (prolactin) was 7 ng/mL and both were between normal ranges. Total testosterone level (416,09 ng/dL) and estradiol level was 34.11 pg/mL (0 - 32,2 pg/mL for postmenopausal women) were increased. Free testesteron was increased (22 nmol/L) and 17-α hydroxyprogesterone was 0,61 ng/mL. Cortisol after 1mg-dexamethasone suppression test (1-mg DST) was 0.65 ug/dl.
In March 2021, the patient underwent a pelvic ultrasound, which was inconclusive due to obesity. However, no masses were detected in the ovaries. An abdominal MR was also performed, revealing no significant findings in the ovaries. She was prescribed spironolactone 100 mg per day and orlistat for weight loss. In November 2021, the patient underwent surgery due to the discovery of a left-sided sex cord-stromal tumor during the pelvic examination. The histopathological examination confirmed the diagnosis of "Luteinized Thecoma" in the left ovary. The tumor was composed of luteinized cells capable of producing androgens, explaining the patient's hirsutism. Following the surgery, the patient experienced a reduction in hirsutism, possibly due to the removal of the hormonally active tumor. Moreover, she exhibited weight loss since the last prescription, with her weight reducing to 98.3 kg, resulting in a BMI of 41.4.

Conclusion
Hyperthecosis is mostly reported in postmenopausal women, affecting less than 1% of women of reproductive age. This case illustrates the diagnostic journey of a postmenopausal woman with hirsutism. The discovery of a Luteinized Thecoma, a rare sex cord-stromal tumor, explains the patient's hirsutism.
P-40 A rare cause of male infertility: 46, XX testicular disorder of sex development
Authors: Erturk Banu , Altan Mesut , Simsek-Kiper Pelin Ozlem , Guvenoglu Merve , Oguz Seda Hanife 
Published: January 2024

Abstract

Introduction
46, XX testicular disorder of sex development (DSD) is a rare condition with an estimated incidence of 1 per 20,000 individuals. The clinical spectrum ranges from ambiguous genitalia that is usually diagnosed at birth, to male infertility that is diagnosed in the adult years. However, the diagnosis of 46, XX DSD in an individual presenting with infertility may be challenging due to occult clinical findings. A thorough physical examination as well as clinical suspicion is required for diagnosis.

Clinical Case
A 22-year-old male presented to the urology clinic for infertility despite at least 3 years of regular unprotected intercourse. On physical examination, his testicles were palpable bilaterally in the scrotum with small volumes. Laboratory results revealed hypergonadotropic hypogonadism and his spermiogram revealed azoospermia. Abdominopelvic ultrosonagraphy (US) was reported normal except abdominal situs inversus. Tumor markers for a probable testicular tumor were negative. He was referred to medical genetic department. His family history was unremarkable except for parental consanguineouty. The chromosomal analyze revealed a 46, XX karyotype, and fluorescence in situ hybridization (FISH) disclosed that the SRY gene locus was translocated to the X chromosome: 46,x,der(x) t(x;Y) (sry+); SRY + AZF a,b,c microdeletion. The patient was consulted to the endocrinology department for further evaluation of hypogonadism. On physical exam, he had eunuchoid habitus with incomplete virilization, bilateral and symmetric gynecomastia. The height, and body mass index of the patient was 165 cm, and 29.2 kg/m2, respectively. The testicles were bilaterally palpable in the scrotum, with a volume of 4 mL each. His luteinizing hormone (LH), follicle-stimulating hormone (FSH) and total testosterone levels were 14 mUI/mL (laboratory reference: 1.5–9 mUI/mL), 42 mUI/mL (1.4–18 mUI/mL), and 140 ng/dl (197-700 ng/dL), respectively. The bone age matched his chronological age. Bone densitometry revealed osteopenia. A direct X-ray of the thorax showed thoracic situs inversus, he diagnosed with situs inversus totalis. Detailed genetic analyses could not be done concerning this diagnosis due to financial constraints. He had no history of lung infections that ruled out Kartagener's syndrome. Treatment was started with monthly intramuscular testosterone injections of 250 mg/ml. The patient and his family received genetic counseling, including advice that assisted reproduction will not work owing to AZFabc deletion.

Conclusion
46, XX DSD is an uncommon genetic disorder that is rarely identified during the evaluation of male infertility. Clinical suspicion and a comprehensive physical exam are keys for diagnosis. Genetic analysis is essential for definitive diagnosis. A multidisciplinary is required for treatment and follow-up. Psychological support and genetic counseling are also critical for the management of these patients.
O-01 Case report: Temozolomide treatment of refractory prolactinoma
Authors: Nekic Anja Barac , Tomsic Karin Zibar , Kastelan Darko , Dusek Tina 
Published: January 2024

Abstract

Introduction
Treatment of refractory prolactinoma is challenging. Temozolomide (TMZ), an oral chemotherapeutic agent, has been recommended as an effective drug for refractory pituitary adenomas or carcinomas.

Clinical Case
We report a 53-year-old male who presented with diplopia, left eye proptosis, conjunctival congestion, and headache. Magnetic resonance imaging (MRI) showed a macroadenoma 20×17 mm in diameter with compression of the optic chiasm and invasion of both cavernous sinuses. Hormonal evaluation revealed hyperprolactinemia of 2382 ug/L, 120x above the upper limit of normal (ULN), with central hypothyroidism and hypogonadism.
Cabergoline was recommended as an initial treatment. Despite the maximum cabergoline dose of 1.5 g daily, tumor volume increased with visual disturbances and headaches. Prolactin (PRL) remained 60x above ULN so the patient underwent transsphenoidal surgery. Tumor histology was consistent with an atypical pituitary adenoma, positively stained for prolactin and sporadically for growth hormone, Ki-67 of 6.4%, no mitosis and negative p53.
Postoperatively, the PRL level was still high (25x above ULN) with tumor remnants in both cavernous sinuses. Due to the tumor resistance to dopamine agonist treatment and uncomplete surgical resection, patient underwent stereotactic gamma knife radiosurgery (GK prescription dose 22 Gy, isodose 49%) simultaneously with TMZ treatment. After 2 cycles of TMZ (1. cycle TMZ 300mg/5days, 2. cycle TMZ 400 mg/5days), the PRL level decreased significantly (126 ug/L). The plan is to apply a total of 6 cycles of temozolomide.

Conclusion
Clinical evidence for the efficacy of TMZ in refractory prolactinoma remains limited, but early use of this agent may be helpful in some cases.
P-65 A case of relapse of Graves ophthalmopathy due to an ophthalmic herpes zoster infection in a thyroidectomized patient with multiple comorbidities
Authors: Radulescu Vlad George , Dumitrascu Anda , Nitu Ileana , Alexandrescu Daniela , Badiu Corin 
Published: January 2024

Abstract

Introduction
Graves ophthalmopathy usually occurs in patients with hyperthyroidism or a history of Graves disease, but it can appear in euthyroid patients or patients with chronic thyroiditis also. It is a possible cause of sight-threatening eye disease. Its incidence is 16 for women and 3 cases for men, per 100 000 population but more severe in men, and it's higher among smokers. In mild forms, treatment can be local, but in severe cases and recurrences, systemic glucocorticoids are compulsory, with or without immunosuppressive drugs, surgery or radiotherapy. in recurrences.

Clinical Case
We present the case of a 76-year-old male, former smoker, diagnosed with Graves disease in 2017. He was treated with anti-thyroid treatment and in 2018 he was thyroidectomized. He also developed a progressive Graves ophthalmopathy from April to July 2017 with a reduced visual acuity for both eyes, conjunctival erythema, palpebral edema and erythema. He received methyl-prednisolone (MP) pulse therapy 6×125 mg weekly with favorable outcome. In 2019, he underwent a recession of internal rectus and inferior rectus for both eyes to correct a divergent strabism and his eye-sight improved. He presents now for an aggravation of the ophthalmopathy after an episode of a herpes zoster infection affecting the ophthalmic branch of the trigeminal nerve. He also suffers from heart failure NYHA III, permanent atrial fibrillation, large aortic stenosis, asymptomatic hyperuricemia, secondary pulmonary hypertension and chronic kidney disease stage IIIb. His medical history revealed hypercholesterolemia, hyperuricemia, heart failure, insufficient status of vitamin D3 and confirmed the chronic kidney disease stage IIIb; normal thyroid function but with high anti TSH receptor antibodies (TRAb=18.37 U/L). Given the age and the comorbidities but also the high activity of the Graves disease and the orbital CT that revealinged a great hypertrophy of the ocular muscles, our ophthalmologist suggested methyl-prednisolone pulse therapy in a standard regimen, but our cardiologist contradicted it because of the heart conditions. As the patient's left eye sight was only 2% and for the right one 20%, we decided to adapt the doses of the pulse therapy and closely monitor the patient. The first dose was 125mg MP, after that 7 doses of 250mg MP weekly was followed by 3 doses of 125mg MP weekly. After a total of 2250 mg MP at a 3 months follow-up, the patients’ eyesight improved to 60% on the left eye and 75% on the right eye and a lower level of TRAb=5.59 UI/L.

Conclusion
Even in patients with comorbidities where corticosteroid therapy may be detrimental, there is a possibility to adapt the doses in a safe way and with a continuous clinical monitoring so that the patients’ eyesight can be saved, therefore increasing the quality of life.
P-69 Beneficial outcomes of hypothyroidism treatment in atrial fibrillation patient
Authors: Abbasova Leyla , Ismayilova Natavan , Huseynova Aygun 
Published: January 2024

Abstract

Introduction
It is well established that hyperthyroidism is a strong independent risk factor for atrial fibrillation (AF). However, the relationship between hypothyroidism and AF is complex and not fully understood. The abnormal thyroid hormone levels can disrupt the electrical signals that regulate heart rhythm, potentially leading to AF. Some symptoms of hypothyroidism, such as fatigue, weakness, and shortness of breath, can mimic the symptoms of AF. This can make it challenging to diagnose AF in individuals with underlying thyroid issues. Meanwhile the treatment of hypothyroidism in A patient with AF can significantly improve the symptoms.

Clinical Case
A 54 years old female was referred to the Therapy department with fatigue and palpitations. She was diagnosed one year ago with paroxysmal tachysystolic atrial fibrillation. CHADSVASK-1, EHRA-3. She experienced paroxysmal episodes four to five times a month for which she took propafenone. At presentation echocardiography (ECHO) showed LVEDD-50, LVEDS-35, IVS-12, LVPW-11, LA-42, EF-57%. The blood sample revealed TSH-8.41 (reference range 0.3-4 mIU/l), free T4-1.15 (reference range 10-25 pmol/l), free T3-3.76 (reference range 2.5-5.8 pmol/l), AbTPO-1040 (reference range 2.5-30 IU/ml). On thyroid ultrasound examination dimensions of right lobe were 18 *23 * 39 mm, left lobe were 22*20*38 mm, isthmus 3.8 mm, and no nodules were present. Total thyroid volume was 19.5 mL. The gland had heterogeneous echotexture and normal vascularity. Treatment with levothyroxine 50 mcg was started. After 6 weeks of treatment with levothyroxine TSH improved to 3.8 mIU/l and patient's fatigue and palpitation symptoms significantly improved, and no new paroxysms were detected. Conclusion: Managing hypothyroidism through thyroid hormone replacement therapy can often help control and sometimes even reverse AF if the arrhythmia was related to thyroid dysfunction. However, some individuals, as in this case, may continue to have AF even after their thyroid levels are normalized. In this case we emphasize the importance of understanding the effect of hypothyroidism on AF.
P-70 Angelman syndrome associated with congenital hypothyroidism and corticotropic insufficiency
Authors: Amine Amani Mohammed El 
Published: January 2024

Abstract

Introduction
Angelman syndrome is a rare neurogenetic disorder by mutation similar to that of Prader-Willi syndrome but of different parental origin. Its association with hypothyroidism is rarely reported in the literature

Clinical Case
We report the case of a 22-month-old infant, followed in neuropediatrics for genetically proven Angelman syndrome and referred to our department for congenital hypothyroidism. Explorations were in favor of an anomaly of hormone synthesis. Hormonal assessment had also revealed corticotropic insufficiency with a small hyophysis on magnetic resonance imaging. The evolution was marked by stability under hydrocortisone and levothyroxine

Conclusion
A single case of association of hypothyroidism and Angelman syndrome has been reported in the literature without a suggested mechanism. In the current state of our knowledge, no association of Angelman syndrome, hypothyroidism and corticotropic insufficiency has been described, making our observation the first to present these three pathologies. The simple association is plausible in our case but the possible description of similar situations in the future would support the hypothesis of a causal relationship.
P-79 Graves’ Disease associated with alemtuzumab use for multiple sclerosis
Authors: Basmacı Nergis , Ünlütürk Uğur 
Published: January 2024

Abstract

Introduction
Alemtuzumab was the first humanized monoclonal antibody to be created and is directed against CD52, a lymphocyte cell surface marker of unknown function. It has been used to treat leukemia and organ transplant rejection. More recently, alemtuzumab has been demonstrated to be highly effective in treating relapsing-remitting multiple sclerosis (MS) disease. Thyroid disease develops in approximately 20% of patients using alemtuzumab for MS, and Graves’ disease is the most common among them. Graves’ disease has not been reported in the use of alemtuzumab for reasons other than MS.

Clinical Case
A 28-year-old female patient with a 10 year history of MS had been treated with pulse corticosteroid, β-interferon and fingolimod. She received alemtuzumab in May 2019 due to increased relapse rates. After the first dose of alemtuzumab, she could not continue her treatment program due to the COVID-19 pandemic. Thyrotoxic thyroid functions (TSH: &lt;0.015 uIU/mL, free T4: 24.79 pmol/L) were detected in the laboratory examination of the patient who was admitted to the endocrinology outpatient clinic with the complaint of palpitation in April 2021. The review of the patient's past laboratory test results determined she was euthyroid before alemtuzumab treatment. She had no personal or family history of thyroid disease. She had no signs and symptoms of thyroid ophthalmopathy.
Ultrasonographic examination of her thyroid gland revealed normal size gland and pseudonodular appearance in the parenchyma. Subsequent thyroid scintigraphy showed diffuse uptake of the radioisotope and reduced counting time of the examination. These results were consistent with Graves’ disease. The patient was started on 5 mg methimazole twice a day. Since the etiology of Graves’ disease was thought to be primarily related to alemtuzumab, the patient's MS treatment was continued with ocrelizumab. She stopped the methimazole treatment within 3 months and did not regularly show up to her follow-up visit. In November 2022, she was admitted to the outpatient department with palpitation, sweating, and irritability symptoms. Thyrotoxic thyroid functions were detected again (TSH:&lt;0,015 uIU/mL, free T4: 26,82 pmol/L). She was restarted on 5 mg methimazole treatment twice a day. In June 2023, she was euthyroid (TSH: 0,496 uIU/mL, free T4: 5,42 pmol/L, free T4: 10,45 pmol/L) and her TSI level was undetectable. The methimazole was reduced to 5 mg once a day. She continues to her treatment and follow-up in our center.

Conclusion
Graves’ disease is a common complication of alemtuzumab use in MS. Alemtuzumab-associated Graves’ disease follows an atypical course compared to the classical form of the disease. The spontaneous remission rate is less than in the classical form of the disease, and medical treatment is often required. Thyroidectomy may be necessary in cases that do not respond to antithyroid agents. Close monitoring of thyroid function tests is essential in alemtuzumab-associated Graves’ disease.
P-83 Primary thyroid lymphoma presented with hypercalcemia and rapidly growing neck mass
Authors: Keser Ali , Öner Hatice , Yavuz Dilek Gogas 
Published: January 2024

Abstract

Introduction
Primary thyroid lymphoma is an uncommon malignancy, accounting for 1% to 2% of extranodal lymphomas and 1% to 5% of thyroid malignancies. Large B-cell lymphoma is the most prevalent form of primary thyroid lymphoma. Here, we present a patient with hypercalcemia and a rapidly growing neck lesion who was diagnosed with diffuse large B-cell lymphoma of the thyroid gland.

Clinical Case
A 75-year-old patient presented to the emergency room with loss of appetite and decreased oral intake over the previous five days, as well as abdominal pain and inability to pass gas and feces. The nausea and vomiting symptoms began simultaneously with the abdominal discomfort. Inspection revealed a distinct, palpable, and erythematous mass in the patient's neck, which the patient reported developing within the previous three months. After noticing the growing mass and consulting her primary care physician, she was referred to an ENT specialist; a biopsy was conducted and the results were pending. Her preliminary laboratory results revealed hypercalcemia and renal failure with depressed parathyroid hormone levels. The following were the initial laboratory results: creatinine 2,14 mg/dL, Ca 13,5 mg/dL, Albumin: 4,2 g/dL, P 2,9 mg/dL, Mg:1 mg/dl, parathormone 11.6 ng/dL procalcitonin 0.04% ng/mL and hemoglobin 10.2% g/dl. Abdominal CT revealed a contracted gallbladder with stone formations in the lumen, but no other abnormalities. The patient was admitted to the internal medicine ward with an initial diagnosis of hypercalcemia and prerenal acute kidney failure as a result of decreased oral intake. The patient's hypercalcemia was determined to be PTH-independent, and malignancy was suspected due to a 4-month history of progressive neck lesion. The thyroid gland was 49×40×87mm in the right and 38×40×62mm in the left, and the isthmus was 50mm thick with lobulated outlines on contrast enhanced cervical MR. The signal intensity of parenchyma was heterogeneous. 18F-FDG PET/CT uncovered a bilateral, distinct increase in the size of the thyroid gland with protrusions to the hyoid bone in superior sequences. The solid mass was found to have diffusely increased FDG uptake and a hypermetabolic pattern, both of which are indicative of malignant characteristics. The lesions encompass the trachea and there are reports of invasion of the cricoid cartilage and thyroid cartilage. A second main malignant disease concern was raised when small intestine FDG uptake and wall thickening suggested malignancy. The patient was re-evaluated by the endocrinology clinic, and additional workup tests were ordered due to the suspicion of Riedel Struma, which was based on the control sedimentation rate of 93 mm/h and the firmness of the gland upon palpation. An open surgical biopsy was performed. The pathology report concluded that it is consistent with diffuse large B-cell lymphoma that is CD20-positive. The hematology department has scheduled chemotherapy treatment for the patient.
P-85 Inflammatory myofibroblastic tumor: An extremely rare tumor of the thyroid gland poses challenges in diagnosis and management
Authors: Nasrullayeva Fidan , Durak Merih Güray , Özbek Süha Süreyya , Makay Özer , Akıncı Barış 
Published: January 2024

Abstract

Introduction
Inflammatory myofibroblastic tumor (IMT) is an exceedingly rare mesenchymal tumor characterized by myofibroblastic spindle cells and an inflammatory cell infiltrate. The clinical presentation of IMT in the thyroid gland varies widely, ranging from an asymptomatic thyroid nodule to a diffuse, rigid, fixed, and painful mass. While IMT of the thyroid gland is generally considered a benign borderline tumor with very low metastatic potential, its diagnosis and management can be intricate. Surgery remains the preferred treatment, with recurrence being rare following successful removal. Nonetheless, the optimal management of residual tumors is contentious and may involve a second surgery or the use of immunosuppressants and chemotherapeutic agents.

Clinical Case
A 47-year-old female patient presented with painless discomfort and stiffness around the thyroid gland. A neck ultrasound revealed a hypoechoic nodule measuring 21×12 mm on the left lobe and a hypoisoechoic heterogeneous nodule 35×28 mm in size on the right lobe of the thyroid gland. These nodules displayed low vascularity, and no pathologic lymph nodes were detected. Fine needle aspiration biopsies (FNABs) were performed on the nodules in both lobes at different times, indicating thyroiditis in the right lobe and spindle-shaped cells with atypia of uncertain significance in the left lobe. Subsequent evaluation of FNAB materials at a referral center suggested a high suspicion of lymphoid malignancy. The patient underwent thyroidectomy; however, the surgeon chose to preserve the left thyroid lobe due to its extreme fibrotic and stone-like consistency, thereby preserving recurrent laryngeal nerve function. Histological analysis confirmed the presence of an inflammatory myofibroblastic tumor characterized by spindle cells accompanied by abundant histocytes, plasma cells, and lymphocytes. The tumor cells exhibited positivity for SMA. Postoperative ultrasound revealed a residual tumor approximately 2 cm in size located in the left isthmus and adjacent left lobe, which remained stable over a 6-month follow-up period.

Conclusion
Our case highlights the potential for misdiagnosing IMT of the thyroid gland as malignant thyroid tumors. Furthermore, the management of a residual tumor presents a complex clinical challenge. The surgical team is reluctant to pursue a second surgery due to potential risks to recurrent laryngeal nerve function. In this session, we aim to solicit expert opinions and engage in interactive discussions regarding the management of the residual tumor in this specific case.