Case Reports in Endocrinology The latest articles from Hindawi Publishing Corporation © 2015 , Hindawi Publishing Corporation . All rights reserved. Life-Threatening Hypercalcemia due to Graves’ Disease and Concomitant Adrenal Failure: A Case Report and Review of the Literature Tue, 24 Mar 2015 07:08:48 +0000 A 47-year-old woman presented with the complaints of nausea, vomiting, and weight loss. She had a history of bilateral surrenalectomy due to Cushing’s syndrome. On examination she had tachycardia and orthostatic hypotension. Laboratory examinations revealed hypercalcemia and suppressed parathyroid hormone levels. She also had thyrotoxicosis due to Graves’ disease. The investigations to rule out a malignancy were negative. With steroid, zoledronic acid, and antithyroid drug treatment her symptoms were resolved and calcium level was normalized. This case highlights the importance of recognizing thyrotoxicosis and concomitant adrenal failure as a possible cause of severe hypercalcemia. Hande Mefkure Ozkaya, Fatma Ela Keskin, Ozlem Asmaz Haliloglu, Tugba Elif Senel, and Pinar Kadioglu Copyright © 2015 Hande Mefkure Ozkaya et al. All rights reserved. Thyroid Storm Precipitated by Duodenal Ulcer Perforation Mon, 09 Mar 2015 10:35:04 +0000 Thyroid storm is a rare and life-threatening complication of thyrotoxicosis that requires prompt treatment. Thyroid storm is also known to be associated with precipitating events. The simultaneous treatment of thyroid storm and its precipitant, when they are recognized, in a patient is recommended; otherwise such disorders, including thyroid storm, can exacerbate each other. Here we report the case of a thyroid storm patient (a 55-year-old Japanese male) complicated with a perforated duodenal ulcer. The patient was successfully treated with intensive treatment for thyroid storm and a prompt operation. Although it is believed that peptic ulcer rarely coexists with hyperthyroidism, among patients with thyroid storm, perforation of a peptic ulcer has been reported as one of the causes of fatal outcome. We determined that surgical intervention was required in this patient, reported despite ongoing severe thyrotoxicosis, and reported herein a successful outcome. Shoko Natsuda, Yomi Nakashima, Ichiro Horie, Takao Ando, and Atsushi Kawakami Copyright © 2015 Shoko Natsuda et al. All rights reserved. Warthin-Like Papillary Thyroid Carcinoma Associated with Lymphadenopathy and Hashimoto’s Thyroiditis Tue, 03 Mar 2015 07:58:04 +0000 Defining the histologic variant of thyroid carcinoma is an important clinical implication as their progression, recurrence, aggressiveness, and prognosis differ. Warthin-like variant is one of the rarest histologic variants of papillary thyroid cancer. A 36-year-old female sought consult for assessment of a painless right neck tumor. High-resolution neck ultrasound revealed a right hypoechoic, 1.71 × 1.05 cm thyroid nodule. Ultrasound-guided fine-needle aspiration biopsy report was a Bethesda grade III. Thyroid function tests showed Hashimoto’s thyroiditis. The patient underwent right hemithyroidectomy. Microscopically, the tumor was composed of papillae lined by cells with eosinophilic cytoplasm, nuclear chromatin clearing, grooves, and pseudoinclusions and a characteristic lymphoplasmacytic infiltrate of the papillae cores. Extension into the perithyroidal soft tissue and 3 ipsilateral lymph nodes was found to be positive for cancer. Warthin-like variant is an uncommon and relatively unknown variant of papillary thyroid carcinoma that has been usually associated with an excellent prognosis. Interestingly, BRAF mutations have been reported to be present in up to 75% of the patients. It is frequently associated with Hashimoto’s thyroiditis and presents unique morphological features that make it recognizable on histologic examination. The cytological diagnosis is difficult to assess due to the overlap in its findings with the classical variant and Hashimoto’s thyroiditis. Karla Judith González-Colunga, Abelardo Loya-Solis, Luis Ángel Ceceñas-Falcón, Oralia Barboza-Quintana, and René Rodríguez-Gutiérrez Copyright © 2015 Karla Judith González-Colunga et al. All rights reserved. Autoimmune Polyendocrine Syndrome 3 Onset with Severe Ketoacidosis in a 74-Year-Old Woman Mon, 02 Mar 2015 09:04:33 +0000 Type 1 diabetes mellitus (T1D), autoimmune thyroid disease, and autoimmune gastritis often occur together forming the so-called autoimmune polyendocrine syndrome type 3 (APS3). We here report a clinical case of a 74-year-old woman who presented for the first time with severe hyperglycemia and ketoacidosis diagnosed as T1D. Further clinical investigations revealed concomitant severe hypothyroidism with autoimmune thyroid disease and severe cobalamin deficiency due to chronic atrophic gastritis. The diagnosis of type 1 diabetes mellitus was confirmed by the detection of autoantibodies against glutamic acid decarboxylase 65, islet cell antibodies, and anti-insulin autoantibodies. Anti-thyroperoxidase, anti-thyroglobulin, and anti-gastric parietal cell antibodies were also clearly positive. The case emphasized that new onset diabetic ketoacidosis, hypothyroidism, and cobalamin deficiency may simultaneously occur, and one disease can mask the features of the other, thereby making diagnosis difficult. It is noteworthy that an APS3 acute episode occurred in an asymptomatic elder woman for any autoimmune diseases. Stefano Benedini, Antonietta Tufano, Elena Passeri, Marco Mendola, Livio Luzi, and Sabrina Corbetta Copyright © 2015 Stefano Benedini et al. All rights reserved. Methimazole Associated Neutropenia in a Preterm Neonate Treated for Hyperthyroidism Tue, 24 Feb 2015 12:06:08 +0000 Maternal Graves’ disease is relatively uncommon with an estimated incidence of 0.4%–1% of all pregnancies, but only 1–5% of newborns delivered to mothers with Graves’ disease develop overt clinical signs and symptoms of hyperthyroidism. Here, we describe a case of a 1380-gram female neonate who was born at 30-week gestation to a mother with Graves’ disease. Our patient presented with hyperthyroidism followed by transient hypothyroidism requiring treatment with levothyroxine. While hyperthyroid, she was treated with methimazole, iodine, and a beta-blocker. 20 days after the initiation of methimazole, she developed neutropenia. The neutrophil counts started to improve immediately after the initiation of the weaning of methimazole. To the best of our knowledge, this is the first case reported in the literature of methimazole induced neutropenia in a preterm infant being treated for neonatal Graves’ disease. Dimitrios Angelis, Rita Ann Kubicky, and Alan B. Zubrow Copyright © 2015 Dimitrios Angelis et al. All rights reserved. Complete Remission of Anaplastic Thyroid Carcinoma after Concomitant Treatment with Docetaxel and Radiotherapy Wed, 18 Feb 2015 13:20:57 +0000 Anaplastic thyroid carcinoma (ATC) although rare is the most lethal form of thyroid cancer. The mortality rate for ATC is very high, with a median survival time of only 5 months; the survival rate at 1 year after diagnosis is <20%. Management of ATC is extremely difficult and rife with uncertainties. Herein, we describe a 75-year-old woman who presented with ATC and was successfully treated using concomitant treatment with docetaxel and high-dose radiotherapy. This case appears to be the first to have been reported in the literature involving complete remission of ATC confirmed by autopsy, suggesting the therapeutic potential of this combination. Ichiro Abe, Satoko Karasaki, Yayoi Matsuda, Shohei Sakamoto, Torahiko Nakashima, Hidetaka Yamamoto, Hisaya Kawate, Keizo Ohnaka, Hisashi Nakashima, Kunihisa Kobayashi, Yoshinao Oda, Masatoshi Nomura, and Ryoichi Takayanagi Copyright © 2015 Ichiro Abe et al. All rights reserved. Case Reports That Illustrate the Efficacy of SGLT2 Inhibitors in the Type 1 Diabetic Patient Mon, 16 Feb 2015 06:49:17 +0000 SGLT2 inhibitors are only approved for use in adults with type 2 diabetes. However, because SGLT2 inhibitors have a mechanism of action that does not require the presence of endogenous insulin, these drugs should also be efficacious in type 1 diabetes where endogenous insulin production is greatly reduced or absent. Herein, I present five cases which illustrate the benefits of utilizing an SGLT2 inhibitor with type 1 diabetes. In these cases the use of SGLT2 inhibitors resulted not only in better glycemic control in most patients but also in some patients’ less hypoglycemia, weight loss, and decreased doses of insulin. In type 1 diabetes Candida albicans vaginitis and balanitis may occur more frequently than in type 2 diabetes. These cases show that a large randomized clinical trial of SGLT2 inhibitors in type 1 diabetes needs to be performed. David S. H. Bell Copyright © 2015 David S. H. Bell. All rights reserved. Anticytotoxic T-Lymphocyte Antigen-4 Induced Autoimmune Hypophysitis: A Case Report and Literature Review Wed, 28 Jan 2015 12:11:12 +0000 Objective. We describe a case of autoimmune hypophysitis induced by the anticytotoxic T-lymphocyte antigen-4 (CTLA-4) agent, ipilimumab. Methods. Case presentation and review of the literature. Results. Autoimmune hypophysitis, a previously described rare disorder, is being recognized more frequently as a side effect of novel immunomodulatory agents used in the treatment of malignancies such as melanoma. CTLA-4 agents are associated with immune-related adverse effects (irAE) which occur as a result of activation (or lack of inactivation) of the immune response. This impacts not only malignant cells but also different host organ-systems. Autoimmune hypophysitis is one of several endocrinopathies associated with these agents. Conclusion. It is important that endocrinologists become familiar with the endocrinopathies, such as autoimmune hypophysitis, associated with new immunomodulator agents which are being used with increasing frequency to treat a variety of malignancies. Deborah Majchel and Mary T. Korytkowski Copyright © 2015 Deborah Majchel and Mary T. Korytkowski. All rights reserved. Central Giant Cell Granuloma of Posterior Maxilla: First Expression of Primary Hyperparathyroidism Mon, 26 Jan 2015 14:04:05 +0000 A case of 19-year-old male patient reported with the chief complaint of slowly growing diffuse painless swelling over the right part of the face from last 6 months. Intraoral examination revealed a swelling on right side of palate in relation to molar region with buccal cortical plate expansion. Radiographic examination (orthopantograph and 3DCT) showed large multilocular radiolucency in right maxilla with generalized loss of lamina dura. Incisional biopsy was done and specimen was sent for histopathological examination which showed multinucleated giant cells containing 15–30 nuclei. Based on clinical, radiological, and histopathological findings provisional diagnosis of central giant cell granuloma was made. Blood tests after histopathology demonstrated elevated serum calcium level and alkaline phosphatase level. Immunoassay of parathyroid hormone (PTH) level was found to be highly elevated. Radiographic examination of long bones like humerus and femur, mandible, and skull was also done which showed osteoclastic lesions. Considering the clinical, radiographic, histopathological, and blood investigation findings, final diagnosis of brown tumour of maxilla was made. The patient underwent partial parathyroidectomy under general anaesthesia to control primary hyperparathyroidism. Surgical removal of the bony lesion was done by curettage. The patient has been followed up for 1 year with no postoperative complications and the lesion healed uneventfully. Deepanshu Gulati, Vishal Bansal, Prajesh Dubey, Sanjay Pandey, and Abhinav Agrawal Copyright © 2015 Deepanshu Gulati et al. All rights reserved. Thyroid Carcinoma with Pituitary Metastases: 2 Case Reports and Literature Review Wed, 21 Jan 2015 11:19:47 +0000 We present 2 patients with pituitary metastases from thyroid carcinoma—the first from anaplastic thyroid carcinoma and the second from follicular thyroid carcinoma. The first patient, a 50-year-old lady, presented with 2-week history of hoarseness of voice, dysphagia, dyspnoea, and neck swelling. Imaging revealed metastatic thyroid cancer to lymph nodes and bone. Histology from surgery confirmed anaplastic thyroid cancer. She was found to have pituitary metastases postoperatively when she presented with nonvertiginous dizziness. She subsequently underwent radiotherapy and radioiodine treatment but passed away from complications. The second patient, a 65-year-old lady, presented with loss of appetite and weight with increased goitre size and dyspnoea. Surgery was performed in view of compressive symptoms and histology confirmed follicular thyroid carcinoma. Imaging revealed metastases to bone, lung, and pituitary. She also had panhypopituitarism with hyperprolactinemia and diabetes insipidus. She received radioiodine therapy but eventually passed away from complications. Weiying Lim, Dawn Shaoting Lim, Chiaw Ling Chng, and Adoree Yiying Lim Copyright © 2015 Weiying Lim et al. All rights reserved. A Case of Type 2 Amiodarone-Induced Thyrotoxicosis That Underwent Total Thyroidectomy under High-Dose Steroid Administration Tue, 13 Jan 2015 10:29:40 +0000 Amiodarone is used commonly and effectively in the treatment of arrhythmia; however, it may cause thyrotoxicosis categorized into two types: iodine-induced hyperthyroidism (type 1 amiodarone-induced thyrotoxicosis (AIT)) and destructive thyroiditis (type 2 AIT). We experienced a case of type 2 AIT, in which high-dose steroid was administered intravenously, and we finally decided to perform total thyroidectomy, resulting in a complete cure of the AIT. Even though steroid had been administered to the patient (maximum 80 mg of prednisolone), the operation was performed safely and no acute adrenal crisis as steroid withdrawal syndrome was found after the operation. Few cases of type 2 AIT that underwent total thyroidectomy with high-dose steroid administration have been reported. The current case suggests that total thyroidectomy should be taken into consideration for patients with AIT who cannot be controlled by medical treatment and even in those under high-dose steroid administration. Koshi Hashimoto, Masaki Ota, Tadanobu Irie, Daisuke Takata, Tadashi Nakajima, Yoshiaki Kaneko, Yuko Tanaka, Shunichi Matsumoto, Yasuyo Nakajima, Masahiko Kurabayashi, Tetsunari Oyama, Izumi Takeyoshi, Masatomo Mori, and Masanobu Yamada Copyright © 2015 Koshi Hashimoto et al. All rights reserved. Late-Onset Metastasis of Renal Cell Carcinoma into a Hot Thyroid Nodule: An Uncommon Finding Not to Be Overlooked Mon, 05 Jan 2015 14:23:53 +0000 We report the case of a 74-year-old man with a four-year history of right nephrectomy for clear cell renal carcinoma (CCRC) who was diagnosed with hyperthyroidism. On ultrasound (US), a 5 cm solid isohypoechoic nodule with intranodular vascularization was found in the left thyroid lobe. The nodule was deemed autonomous on thyroid scan. Methimazole was started and serum thyroid hormone levels quickly normalized; euthyroidism was maintained with a very low dosage of antithyroid drug. Over time, compressive symptoms and local pain occurred and US revealed growth of the nodule. Total thyroidectomy was performed and the combined histological and immunohistochemical evaluation deemed the nodule compatible with metastasis of CCRC; on 2-year follow-up, no tumor relapse was ascertained. In patients with a history of cancer, a thyroid nodule, even if hyperfunctioning, must be suspected of being a metastasis and investigated. Hot nodules, which are largely benign, may be vulnerable to metastatic colonization owing to their rich vascularization. In these cases, surgery may be curative. Luca Foppiani, Michela Massollo, Patrizia Del Monte, Roberto Bandelloni, Anselmo Arlandini, and Arnoldo Piccardo Copyright © 2015 Luca Foppiani et al. All rights reserved. A Case of Primary Hyperparathyroidism due to Intrathyroidal Parathyroid Cyst Mon, 29 Dec 2014 10:59:30 +0000 Parathyroid cysts constitute 0.08–3.41% of all parathyroid masses. Intrathyroidal parathyroid cysts, however, are rare conditions with only a few cases being reported. Most of the parathyroid cysts are found to be nonfunctional and functional cysts are generally thought to be due to cystic degeneration of parathyroid adenomas. A cystic, smooth contoured lesion of 24 × 19 × 16 mm was observed in left thyroid lobe of a 76-year-old woman during ultrasonography which was performed as routine workup for primary hyperparathyroidism. It was defined as a cystic thyroid nodule at first. Tc99m sestamibi scintigraphy was performed to see any parathyroid lesions, but no radioactive uptake was observed. Intact parathormone (iPTH) level was found to be >600 pg/mL in cyst aspiration fluid. Left lobectomy was performed, with a diagnosis of primary hyperparathyroidism due to functional parathyroid cyst. Serum iPTH level was decreased >50% postoperatively and histopathological evaluation was consistent with an encapsulated parathyroid adenoma with a cystic center. Parathyroid cysts are among rare causes of primary hyperparathyroidism. Diagnosis is made by markedly increased iPTH level in cyst fluid and observation of parathyroid epithelium lining the cyst wall. Yavuz Yalcin, Turkan Mete, Recep Aktimur, Gultekin Ozan Kucuk, Gulhan Duman, Aysu Basak Ozbalci, and Omer Alici Copyright © 2014 Yavuz Yalcin et al. All rights reserved. Spontaneous Regression of a Carcinoid Tumor following Pregnancy Sun, 21 Dec 2014 11:30:58 +0000 We present a case of spontaneous regression of a neuroendocrine tumor following pregnancy in the absence of chemotherapy, radiotherapy, or alternative medicine (including herbal medicine). The diagnosis of a nonsecretory carcinoid tumor was confirmed using CT imaging, octreotide scan, and histology. Furthermore, serial imaging has demonstrated spontaneous regression of the carcinoid suggesting that pregnancy did not worsen the course of the disease but instead may have contributed to tumour regression. We discuss mechanisms underlying tumour regression and the possible effect of pregnancy on these processes. A. Sewpaul, D. Bargiela, A. James, S. J. Johnson, and J. J. French Copyright © 2014 A. Sewpaul et al. All rights reserved. An Interesting Cause of Hyperandrogenemic Hirsutism Tue, 16 Dec 2014 11:52:36 +0000 Mild clinical signs of hyperandrogenism such as hirsutism may appear during the menopausal transition as part of the normal aging process, but the development of frank virilization suggests a specific source of androgen excess. We report a case of a 68-year-old woman with signs of virilization that had started 6 months before. Clinical analyses revealed high levels of serum testosterone for a postmenopausal woman. Pelvic MRI and abdomen CT showed no evidence of ovarian and adrenal tumor. Postmenopausal hyperandrogenism can be the result of numerous etiologies ranging from normal physiologic changes to ovarian or rarely adrenal tumors. Our patient was found to have iatrogenic hyperandrogenism. This condition is rarely reported cause of virilization. Murat Atmaca, İsmet Seven, Rıfkı Üçler, Murat Alay, Veysi Barut, Yaren Dirik, and Yasin Sezgin Copyright © 2014 Murat Atmaca et al. All rights reserved. Cutaneous Sinus Formation Is a Rare Complication of Thyroid Fine Needle Aspiration Biopsy Mon, 08 Dec 2014 10:00:55 +0000 Fine needle aspiration biopsy (FNAB) is essential in the diagnosis and management of thyroid nodules. In this paper, we report a rare complication, cutaneous sinus formation, after diagnostic FNAB guided by palpation. Sixty-three-year-old female patient was admitted with the complaints of hoarseness and discharge from the anterior neck wall which were present for the last 6 months. The patient underwent a near total thyroidectomy 17 years ago. Recurrent nodular goiter was detected six months before and a diagnostic FNAB guided by palpation was performed. Two weeks later the patient had wound discharge and hoarseness. Physical examination of the patient revealed a sinus, which was located superior to the thyroidectomy incision. A 1 cm nodule was palpated in the left side of her neck. A cervical ultrasonography (USG) showed a 9 × 7 mm nodule in the remnant thyroid and a 9.5 × 3.5 mm fistulized fluid collection. The patient underwent sinus tract and remnant thyroid removal. This case report presents a cutaneous sinus formation deriving from the granulation tissue, probably due to the silk suture reaction in the previous surgery, by the FNAB guided by palpation procedure. We suggest USG guided FNAB to achieve more accurate and safe diagnosis in evaluating the thyroid nodules. Gülhan Akbaba, Muhyettin Omar, Murat Polat, Önder Özcan, Ahmet Korkut Bellı, Murat Şahan, and Neşat Çullu Copyright © 2014 Gülhan Akbaba et al. All rights reserved. A Pregnant Woman Who Underwent Laparoscopic Adrenalectomy due to Cushing’s Syndrome Wed, 03 Dec 2014 06:38:37 +0000 Cushing’s syndrome (CS) may lead to severe maternal and fetal morbidities and even mortalities in pregnancy. However, pregnancy complicates the diagnosis and treatment of CS. This study describes a 26-year-old pregnant woman admitted with hypertension-induced headache. Hormonal analyses performed due to her cushingoid phenotype revealed a diagnosis of adrenocorticotropic hormone- (ACTH-) independent CS. MRI showed a 3.5 cm adenoma in her right adrenal gland. After preoperative metyrapone therapy, she underwent a successful unilateral laparoscopic adrenalectomy at 14-week gestation. Although she had a temporary postoperative adrenal insufficiency, hormonal analyses showed that she has been in remission since delivery. Findings in this patient, as well as those in previous patients, indicate that pregnancy is not an absolute contraindication for laparoscopic adrenalectomy. Rather, such surgery should be considered a safe and efficient treatment method for pregnant women with cortisol-secreting adrenal adenomas. Halit Diri, Fahri Bayram, Yasin Simsek, Yusuf Ozkan, Alper Akcan, Ibrahim Karahan, Ibrahim Ileri, Sulbiye Aribas, and Mehmet Sait Koc Copyright © 2014 Halit Diri et al. All rights reserved. Hypochondroplasia, Acanthosis Nigricans, and Insulin Resistance in a Child with FGFR3 Mutation: Is It Just an Association? Wed, 19 Nov 2014 00:00:00 +0000 FGFR3 mutations cause wide spectrum of disorders ranging from skeletal dysplasias (hypochondroplasia, achondroplasia, and thanatophoric dysplasia), benign skin tumors (epidermal nevi, seborrhaeic keratosis, and acanthosis nigricans), and epithelial malignancies (multiple myeloma and prostate and bladder carcinoma). Hypochondroplasia is the most common type of short-limb dwarfism in children resulting from fibroblast growth factor receptor 3 (FGFR3) mutation. Acanthosis nigricans might be seen in severe skeletal dysplasia, including thanatophoric dysplasia and SADDAN syndrome, without a biochemical evidence of hyperinsulinemia. Insulin insensitivity and acanthosis nigricans are uncommonly seen in hypochondroplasia patients with FGFR3 mutations which may represent a new association. We aim to describe the association of hypochondroplasia, acanthosis nigricans, and insulin resistance in a child harboring FGFR3 mutation. To our knowledge, this is the first case report associating the p.N540 with acanthosis nigricans and the second to describe hyperinsulinemia in hypochondroplasia. This finding demonstrates the possible coexistence of insulin insensitivity and acanthosis nigricans in hypochondroplasia patients. Manal Mustafa, Nabil Moghrabi, and Bassam Bin-Abbas Copyright © 2014 Manal Mustafa et al. All rights reserved. Bullosis Diabeticorum: Rare Presentation in a Common Disease Tue, 18 Nov 2014 09:33:58 +0000 A 27-year-old African American male presented with a sudden onset of blisters. He had a past medical history of uncontrolled diabetes mellitus type I, diabetic vasculopathy, and neuropathy. The physical examination revealed nonerythematous skin denudations on both elbows and lateral aspect of arm bilaterally. Investigations which included skin biopsies confirmed the diagnosis of bullosis diabeticorum. The bullae were treated with hydrotherapy and healed with no complications in 4 weeks. We present this case to illustrate the rare occurrence of diabetic bulla in a diabetic patient especially with poor glycemic control. The case is also a reminder of the importance of diabetes screening in nondiabetic patients who are diagnosed with diabetic bulla. Vineet Gupta, Neha Gulati, Jaya Bahl, Jaswinder Bajwa, and Naveen Dhawan Copyright © 2014 Vineet Gupta et al. All rights reserved. Potentially Life-Threatening Phosphate Diabetes Induced by Ferric Carboxymaltose Injection: A Case Report and Review of the Literature Thu, 13 Nov 2014 00:00:00 +0000 We report the case of a 45-year-old female patient who developed phosphate diabetes after administration of ferric carboxymaltose. Ten days after the second dose, she complained of intense fatigue and blood analysis showed a phosphate plasma level of 0.93 mg/dL with phosphate excretion rate of 23%. She received phosphate supplementation which resulted in phosphate clearance improvement which persisted for two months. We reviewed other cases described in the literature and would draw attention to this rare but potentially life-threatening side effect. Xavier Vandemergel and Frédéric Vandergheynst Copyright © 2014 Xavier Vandemergel and Frédéric Vandergheynst. All rights reserved. A Case of Rathke’s Cleft Cyst Associated with Transient Central Adrenal Insufficiency and Masked Diabetes Insipidus Thu, 06 Nov 2014 13:45:46 +0000 A 73-year-old woman admitted to our hospital because of headache, poor appetite, malaise, weight loss, and vomiting was found to have central adrenal insufficiency and thyrotoxicosis due to silent thyroiditis. Polyuria developed after replacement with glucocorticoid (masked diabetes insipidus), which was controlled with nasal administration of desmopressin. Magnetic resonance imaging of the brain showed a large cystic pituitary mass (18 × 18 × 12 mm) extending suprasellarly to the optic chiasm. Transsphenoidal surgery revealed that the pituitary tumor was Rathke’s cleft cyst. Following surgery, replacement with neither glucocorticoid nor desmopressin was needed any more. Therefore, it is suggested that Rathke’s cleft cyst is responsible for the masked diabetes insipidus and the central insufficiency. Furthermore, it is speculated that thyrotoxicosis with painless thyroiditis might induce changes from subclinical adrenal insufficiency to transiently overt insufficiency. Masahiro Asakawa, Rina Chin, Yoshihiro Niitsu, Tetsuo Sekine, Arisa Niwa, Atsuko Miyake, Naoko Inoshita, Mitsunobu Kawamura, Yoshihiro Ogawa, and Yukio Hirata Copyright © 2014 Masahiro Asakawa et al. All rights reserved. Psychiatric Symptoms due to Thyroid Disease in a Female Adolescent Tue, 04 Nov 2014 11:46:20 +0000 The hypothalamic-pituitary-thyroid axis is involved in the production of thyroid hormone which is needed to maintain the normal functioning of various organs and systems, including the central nervous system. This study reports a case of hypothyroidism in a fifteen-year-old female adolescent who was attended for psychiatric symptoms. This case reveals the importance of evaluating thyroid function in children and adolescents with neuropsychiatric symptoms. Nelly Capetillo-Ventura and Inmaculada Baeza Copyright © 2014 Nelly Capetillo-Ventura and Inmaculada Baeza. All rights reserved. Pediatric Cushing’s Disease and Pituitary Incidentaloma: Is This a Real Challenge? Mon, 20 Oct 2014 11:13:15 +0000 Cushing’s disease (CD) is the most common cause of endogenous Cushing’s syndrome in children and adolescents and represents a rare cause of short stature. A 14-year-old boy came to our attention for progressive weight gain and short stature. At examination, height was 140 cm (3rd centile) and weight was 37.7 kg (10th centile). Tanner stage was G2, PH 3, testis 3 mL. Hypothyroidism and growth hormone deficiency were excluded. A marked increase of urinary free cortisol, a nonsuppressible serum cortisol after Liddle 1 test, and an elevated ACTH value confirmed the diagnosis of ACTH dependent Cushing’s syndrome. Pituitary magnetic resonance imaging (MRI) showed a left microadenoma and a right focal area of lesser enhancement. Therefore, bilateral inferior petrosal sinus sampling (BIPSS) with CRH stimulation was performed to obtain an accurate preoperative localization of the adenoma: the interpetrosal sinus ACTH gradient indicated lateralization of ACTH secretion to the left side. The patient underwent transsphenoidal surgery with selective microadenomectomy, with an immediate ACTH decline in the postoperative phase. Histology confirmed the diagnosis of corticotrophic pituitary adenoma. Glucocorticoid replacement therapy was instituted. Clinical examination demonstrated a rapid catch-up growth (10th centile), with a normalization of body mass index and an adequate pubertal development. Rosa Maria Paragliola, Pietro Locantore, Alfredo Pontecorvi, and Salvatore Maria Corsello Copyright © 2014 Rosa Maria Paragliola et al. All rights reserved. Metastatic Follicular Thyroid Carcinoma Secreting Thyroid Hormone and Radioiodine Avid without Stimulation: A Case Report and Literature Review Tue, 07 Oct 2014 10:04:14 +0000 Introduction. This is an extremely rare case of a patient with metastatic follicular thyroid cancer who continued to produce thyroid hormone and was iodine scan positive without stimulation after thyroidectomy and radioiodine (I-131) therapy. Patient Findings. A 76-year-old Caucasian male was diagnosed with metastatic follicular thyroid carcinoma on lung nodule biopsy. Total thyroidectomy was performed and he was ablated with 160 mCi of I-131 after recombinant human thyrotropin (rhTSH) stimulation. Whole body scan (WBS) after treatment showed uptake in bilateral lungs, right sacrum, and pelvis. The thyroglobulin decreased from 2,063 to 965 four months after treatment but rapidly increased to 2,506 eleven months after I-131. Thyroid stimulating hormone (TSH) remained suppressed and free T4 remained elevated after I-131 therapy without thyroid hormone supplementation. He was treated with an additional 209 mCi with WBS findings positive in lung and pelvis. Despite I-131, new metastatic lesions were noted in the left thyroid bed and large destructive lesion to the first cervical vertebrae four months after the second I-131 dose. Conclusions. This case is exceptional because of its rarity and also due to the dissociation between tumor differentiation and aggressiveness. The metastatic lesions continued to secrete thyroid hormone and remained radioiodine avid with rapid progression after I-131 therapy. Syed A. Abid, Brendan C. Stack Jr., and Donald L. Bodenner Copyright © 2014 Syed A. Abid et al. All rights reserved. Pemphigus Vulgaris with Solitary Toxic Thyroid Nodule Sun, 21 Sep 2014 09:39:05 +0000 Background. Pemphigus vulgaris is an autoimmune vesiculobullous disease, affecting the skin and mucous membranes. It is reported to be associated with other autoimmune diseases including autoimmune thyroid diseases. However we report herein a case of pemphigus vulgaris associated with autonomous toxic nodule. Case Presentation. A 51-year-old woman was evaluated for blisters and erosions that develop on her trunk, face, and extremities, with a five-year history of progressively enlarging neck mass, and a past medical history of pemphigus vulgaris seven years ago. The condition was associated with palpitation, dyspnea, and heat intolerance. Thyroid function tests and thyroid scan were compatible with the diagnosis of thyrotoxicosis due to autonomous toxic nodule. Exacerbation of pemphigus vulgaris was proved by skin biopsy from the patient which revealed histologic picture of pemphigus vulgaris. Conclusion. Autoimmune thyroid diseases are reported to associate pemphigus vulgaris. To our knowledge, this case is the first in the English literature to report association between pemphigus vulgaris and autonomous toxic nodule and highlights the possibility of occurrence of pemphigus vulgaris with a nonautoimmune thyroid disease raising the question: is it just a coincidence or is there an explanation for the occurrence of both conditions together? Mostafa Alfishawy, Karim Anwar, Amira Elbendary, and Ahmed Daoud Copyright © 2014 Mostafa Alfishawy et al. All rights reserved. SDHB-Associated Paraganglioma in a Pediatric Patient and Literature Review on Hereditary Pheochromocytoma-Paraganglioma Syndromes Mon, 15 Sep 2014 00:00:00 +0000 Pheochromocytoma and paraganglioma are rare in the pediatric population occurring in approximately 1 in 50,000 children. While some cases are sporadic, they have commonly been associated with syndromes such as von Hippel-Lindau, multiple endocrine neoplasia types IIa and IIb, neurofibromatosis type 1, and hereditary pheochromocytoma-paraganglioma syndromes. In children less than 18 years of age approximately 60% of pheochromocytomas and paragangliomas are associated with a germline mutation. We present an 11-year-old child with an abdominal paraganglioma related to a succinate dehydrogenase subunit B gene mutation whose father had a previously resected abdominal paraganglioma and was found to carry the same mutation. In addition, we review the etiology, genetics, diagnostic approach, and challenges of preoperative management of secretory pheochromocytomas and paragangliomas in children. Heather Choat, Kerri Derrevere, Lisa Knight, Whitney Brown, and Elizabeth H. Mack Copyright © 2014 Heather Choat et al. All rights reserved. Sphenoid Brown Tumor Associated with a Parathyroid Carcinoma Mon, 08 Sep 2014 09:14:47 +0000 Brown tumors are osteolytic bone lesions that occur as a result of persistent hyperparathyroidism. They usually appear late in the natural history of the disease and are currently very rare due to an earlier diagnosis of primary hyperparathyroidism. We present the case of a 53-year-old female with a 2-month history of bitemporal hemianopsia and diplopia. A computed tomography showed an osteolytic bone lesion that involved the sphenoid corpus and clivus. A biopsy was made and the histopathology result was consistent with a brown tumor. The aforementioned location is very infrequent to such tumors, and therefore represented a diagnostic challenge. However, in this case, its association to primary hyperparathyroidism was the clue for the diagnosis. The association of a brown tumor secondary to a parathyroid carcinoma has been seldom reported. This case represents, to our knowledge, one of the few brown tumors described in such location and the first secondary to a parathyroid carcinoma. Abelardo Loya-Solís, Alejandra Mendoza-García, Luis Ceceñas-Falcón, and René Rodríguez-Gutiérrez Copyright © 2014 Abelardo Loya-Solís et al. All rights reserved. A Case of Adenomatous Goiter Involving Diffuse, Acute, and Painful Thyroid Enlargement after Fine-Needle Aspiration Cytology Wed, 03 Sep 2014 12:03:17 +0000 The patient was a 44-year-old woman who exhibited a diffuse goiter during health screening. Her medical history did not include any significant medication-based treatment. An echographic examination detected a solid cystic tumor, which measured 21 × 14 × 10 mm, in her right thyroid lobe; however, she displayed normal thyroid function. After fine-needle aspiration cytology had been performed with a 22 G injection needle, the patient immediately complained of compression and pain extending from the front of her neck to her lower chin, which was not accompanied by dyspnea. A second echographic examination revealed diffuse and edematous enlargement and increased internal blood flow in the bilateral thyroid lobes as well as a thyroid nodule. We immediately iced the patient’s neck and administered 125 mg methylprednisolone via an intravenous infusion. Within one hour, her symptoms had markedly improved, but acute pain remained. Thus, we continued the steroid (prednisone) treatment, but the dose was gradually reduced from 10 mg/day to 5 mg/day at 1 week after the patient’s symptoms disappeared. The mechanism responsible for the patient’s condition remains unclear. Toshiro Shimo, Katsuhiro Tanaka, Ryohei Ogata, Wataru Saito, Yusuke Ohta, Yoshikazu Koike, Tetsumasa Yamashita, Yutaka Yamamoto, and Junichi Kurebayashi Copyright © 2014 Toshiro Shimo et al. All rights reserved. Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin Sun, 24 Aug 2014 07:58:40 +0000 In our case, a 45-year-old male patient had multiple fractures accompanied by hypophosphatemia. FGF-23 levels were significantly increased, and total body magnetic resonance imaging (MRI) revealed a tumor mass located at the distal tibia leading to the diagnosis of tumor-induced osteomalacia (TIO). After resection of the tumor, hypophosphatemia and the increased levels of FGF-23 normalized within a few days. Subsequent microscopic examination and immunohistochemical analysis revealed a phosphaturic mesenchymal tumor mixed connective tissue variant (PMTMCT) showing a positive expression of somatostatin receptor 2A (SSTR2A), CD68, and Periostin. Electron microscopy demonstrated a poorly differentiated mesenchymal tumor with a multifocal giant cell component and evidence of neurosecretory-granules. However, the resected margins showed no tumor-free tissue, and therefore a subsequent postoperative radiotherapy was performed. The patient is still in complete remission after 34 months. Tumor resection of PMTMCTs is the therapy of choice. Subsequent radiotherapy in case of incompletely resected tumors can be an important option to avoid recurrence or metastasis even though this occurs rarely. The prognostic value of expression of Periostin has to be evaluated more precisely in a larger series of patients with TIO. Anke H. Hautmann, Josef Schroeder, Peter Wild, Matthias G. Hautmann, Elisabeth Huber, Patrick Hoffstetter, Martin Fleck, and Christiane Girlich Copyright © 2014 Anke H. Hautmann et al. All rights reserved. Chronic Heroin Dependence Leading to Adrenal Insufficiency Wed, 20 Aug 2014 08:28:11 +0000 Opioids have been the mainstay for pain relief and palliation over a long period of time. They are commonly abused by drug addicts and such dependence usually imparts severe physiologic effects on multiple organ systems. The negative impact of opioids on the endocrine system is poorly understood and often underestimated. We describe a patient who developed severe suppression of the hypothalamic-pituitary adrenal (HPA) axis leading to secondary adrenal insufficiency due to long standing abuse of opioids. Gautam Das Copyright © 2014 Gautam Das. All rights reserved.