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, Samantha Neo, Vivian Auyeung, John Weinman
Published: 1 February 2021
Endocrine Practice, Volume 27, pp 146-151; https://doi.org/10.4158/ep-2020-0543

The publisher has not yet granted permission to display this abstract.
Junke Xia, Furong Liu, Jing Wu, Yanjie Xia, Zhenhua Zhao, Yongjiang Zhao, Huayan Ren, Xiangdong Kong
Published: 1 February 2021
Endocrine Practice, Volume 27, pp 137-145; https://doi.org/10.4158/ep-2020-0478

The publisher has not yet granted permission to display this abstract.
Jianbo Li, Gaofei He, Yifan Tong, Li Tao, Lei Xie, Li Gao, Deguang Zhang
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 15-20; https://doi.org/10.4158/ep-2020-0129

Abstract:
Background: Papillary thyroid carcinoma (PTC) can frequently metastasize to the cervical lymph node, especially in the central compartment. Some surgeons believe that dissection posterior to the right recurrent laryngeal nerve lymph node (PRRLN-LN) is unnecessary because of the low metastasis rate and high complication risk. However, persistent metastatic lymph nodes may have a higher recurrence rate, surgical risk, and complications. Thus, it is important to distinguish patients who require PRRLN-LN dissection. The aim of this study was to identify the risk factors for PRRLN-LN metastasis (LN-prRLN), and to establish a scoring system, to help determine whether PRRLN-LN dissection is required in PTC patients. Methods: The study comprised 821 patients with primary PTC in the right or both lobes who had undergone right lobectomy or total thyroidectomy with only right, or bilateral central compartment dissection with/without lateral neck dissection, between January 2010 and June 2016 in our institution. Participants were randomly allocated to development and validation cohorts in a 2:1 ratio. A nomogram-based predictive model for LN-prRLN was established based on the risk factors identified in the development cohort. Results: LN-prRLN was diagnosed pathologically in 15.1% (124/821) of patients from the entire cohort. Multivariate analysis identified age (odds ratio [OR] 0.964, 95% confidence interval [CI] 0.945–0.983; P<0.001), tumor size (OR 1.536, 95%CI 1.135–2.079; P=0.005), extrathyroidal extension (OR 2.271, 95%CI 1.368–3.770; P=0.002), clinically-involved right central compartment lymph node metastasis (OR 1.643, 95%CI 1.055–2.559; P=0.028), and right lateral lymph node metastasis (OR 4.271, 95%CI 2.325–7.844; P<0.001) as predictors of LN-prRLN. A risk model was established and well validated. Calibration curves to evaluate the nomogram in both the development and validation cohorts revealed a C-index of 0.756±0.058 and 0.745±0.042, respectively. Conclusions: Our scoring system may be useful for helping surgeons to decide which patients should undergo dissection of the PRRLN-LN. Abbreviations: ATA = American Thyroid Association; CCD = Central compartment dissection; CI = Confidence interval; C-index = The concordance index; ETE = Extrathyroidal extension; FNA = Fine-needle aspiration; LNM = Lymph node metastasis; LN-prRLN = Posterior to the right recurrent laryngeal nerve lymph node metastasis; OR = Odds ratio; PRRLN-LN = Posterior to the right recurrent laryngeal nerve lymph node; PTC = Papillary thyroid carcinoma; RLN = Recurrent laryngeal nerve.
Gregory P. Westcott, Christopher M. Mulla, James V. Hennessey
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 8-14; https://doi.org/10.4158/ep-2020-0210

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, Shanlee Davis, Leena Nahata
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 56-62; https://doi.org/10.4158/ep-2020-0287

The publisher has not yet granted permission to display this abstract.
Mrinali Tripathi, April Wong, Victoria Solomon,
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 71-76; https://doi.org/10.4158/ep-2020-0135

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, Tine De Backer, Patrick Calders, Bart Pauwels, Samyah Shadid, Bruno Lapauw
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 44-50; https://doi.org/10.4158/ep-2020-0293

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Bekir Ucan, Muhammed Kizilgul, Alper Cagri Karci, Hakan Duger, Muhammed Erkam Sencar, Narin Nasiroglu Imga, Taner Demirci, Dilek Berker, Erman Cakal
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 51-55; https://doi.org/10.4158/ep-2020-0398

The publisher has not yet granted permission to display this abstract.
, Luciano Colangelo, Chiara Sonato, Marco Occhiuto, Carla Ferrara, Andrea del Fattore, Rachele Santori, Monia Mastrantonio, Alessandro Sgreccia, Salvatore Minisola, et al.
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 21-26; https://doi.org/10.4158/ep-2020-0405

The publisher has not yet granted permission to display this abstract.
Panicha Chantrapanichkul, Mary O. Stevenson, Pichatorn Suppakitjanusant, Michael Goodman,
Published: 1 January 2021
Endocrine Practice, Volume 27, pp 27-33; https://doi.org/10.4158/ep-2020-0414

The publisher has not yet granted permission to display this abstract.
Wen-Bin Zheng, Yi Dai, Jing Hu, Di-Chen Zhao, Ou Wang, Yan Jiang, Wei-Bo Xia, Xiao-Ping Xing, Mei Li
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1477-1485; https://doi.org/10.4158/ep-2020-0073

The publisher has not yet granted permission to display this abstract.
Edy Kornelius, Chien-Ning Huang, Shih-Chang Lo, Yu-Hsun Wang, Yi-Sun Yang
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1486-1496; https://doi.org/10.4158/ep-2020-0143

The publisher has not yet granted permission to display this abstract.
John N. Shumar, Mohamed K.M. Shakir,
Published: 1 December 2020
Endocrine Practice, Volume 26; https://doi.org/10.4158/ep-2020-0385

, Lynda Shey, R. Philip Eaton
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1514-1523; https://doi.org/10.4158/ep-2020-0347

The publisher has not yet granted permission to display this abstract.
Shrinath Shetty, Kripa Elizabeth Cherian, Sahana Shetty, , Felix K. Jebasingh, Anish Cherian, Julie Hephzibah, , Reetu Amirta John, Hesarghatta S. Asha, et al.
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1442-1450; https://doi.org/10.4158/ep-2020-0148

The publisher has not yet granted permission to display this abstract.
Boni Xiang, XiaoMing Zhu, Min He, Wei Wu, HaoPeng Pang, Zhaoyun Zhang, Yehong Yang, Yiming Li, Yongfei Wang, Yang Wang, et al.
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1458-1468; https://doi.org/10.4158/ep-2020-0192

The publisher has not yet granted permission to display this abstract.
Chenxiang Cao, Sina Jasim, Amrita Cherian, Aziza Nassar, Ana-Maria Chindris, Ana Marcella Rivas, Stephanie Bonnett, Melanie Caserta, Marius N. Stan,
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1497-1504; https://doi.org/10.4158/ep-2020-0403

The publisher has not yet granted permission to display this abstract.
Ana Laura Espinosa-De-Los-Monteros, ,
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1435-1441; https://doi.org/10.4158/ep-2020-0368

The publisher has not yet granted permission to display this abstract.
Qian-Hui Xu, Qiu-Yu Li, Kang Yu, Qian-Ming Ge, Wen-Qing Shi, Biao Li, Rong-Bin Liang, Qi Lin, Yu-Qing Zhang, Yi Shao
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1399-1405; https://doi.org/10.4158/ep-2020-0045

The publisher has not yet granted permission to display this abstract.
Thomas Ittoop, Kimberly Jeffrey, Chin-I Cheng,
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1425-1434; https://doi.org/10.4158/ep-2020-0239

The publisher has not yet granted permission to display this abstract.
Jung Hee Kim, Min Joo Kim, Jung Hyun Lee, Ji Won Yoon, Chan Soo Shin
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1406-1415; https://doi.org/10.4158/ep-2020-0182

The publisher has not yet granted permission to display this abstract.
Timothy P. Foster, Brittany Bruggeman, Martha Campbell-Thompson, Mark A. Atkinson, Michael J. Haller, Desmond A. Schatz
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1505-1513; https://doi.org/10.4158/ep-2020-0295

The publisher has not yet granted permission to display this abstract.
, Akira Miyauchi, Yasuhiro Ito, Mitsuru Ito, Takumi Kudo, Mika Tanaka, Kazuyoshi Kohsaka, Toshihiko Kasahara, Eijun Nishihara, Shuji Fukata, et al.
Published: 1 December 2020
Endocrine Practice, Volume 26, pp 1451-1457; https://doi.org/10.4158/ep-2020-0201

The publisher has not yet granted permission to display this abstract.
Ethan I. Fieger, Kristen M. Fadel, Amir H. Modarres, Edmond P. Wickham,
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0314

Abstract:
Objective: We report a case of a successful reimplementation of a very low carbohydrate ketogenic diet (VLCKD) after a case of euglycemic diabetic ketoacidosis (euDKA). Methods: A 42-year-old female with a history of type 2 diabetes mellitus (T2DM) on a self- administered VLCKD was prescribed a sodium-glucose co-transporter 2 (SGLT2) inhibitor. Two weeks after initiation, she presented with nausea and vomiting and was found to be in euDKA which was treated with fluid resuscitation, insulin infusion and cessation of the SGLT2 inhibitor. She was discharged on insulin and instructed not to resume a VLCKD. Results: After discharge, the patient experienced rapid weight gain and deteriorating glycemic control and desired to resume a VLCKD. She was referred to a university-based medical weight loss clinic that specializes in a VLCKD. The patient was monitored with daily contact via the electronic health record’s (EHR) patient portal and serial laboratory testing while her carbohydrate intake was slowly reduced and her insulin titrated off. She has safely remained in ketosis for two years without a further episode of euDKA. Conclusion: As the clinical use of SGLT2 inhibitors and the VLCKD both become increasingly common, it is vital for practitioners to be aware that the combination can lead to euDKA. We present a case of successfully resuming a VLCKD after recovering from euDKA and cessation of SGLT2 inhibitor therapy.
, Henry J. Rohrs
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0103

Abstract:
Objective: Acrodysostosis is a rare skeletal dysplasia with one gene mutation associated with pseudohypoparathyroidism. We describe a 15-year-old male patient with genetically proven acrodysostosis who presents with hyperparathyroidism. Methods: Laboratory testing including genetic testing for acrodysostosis and biochemical evaluation for hypercalcemia were obtained. For evaluation of the source of hyperparathyroidism, parathyroid imaging including sestamibi scan, ultrasound, and 4DCT scan were performed. Results: Initial calcium 11.7 mg/dL (8.4–10.2), phosphorus 2.6 mg/dL (2.9–5.0), and PTH 177 pg/ml (15–65) were suspicious for hyperparathyroidism. Magnesium, albumin, creatinine, and PTHrP levels were normal. His Ca/Cr ratio was 0.15, calcium/creatinine clearance ratio was 0.008 and the fractional excretion of phosphorus was 34%. Our patient had no symptoms other than long standing bone pain. Thyroid ultrasound, then sestamibi scan did not show a parathyroid adenoma nor parathyroid gland hyperplasia. Familial hypocalciuric hypercalcemic syndrome (FHH) was entertained, but without a family history and documented normal calcium levels throughout childhood, FHH was unlikely. On subsequent testing, his calcium and PTH levels increased. Subsequent imaging including repeat thyroid ultrasound, Sestamibi scan, and CT scan have not found a definitive cause. MEN1 genetic testing was negative. Without an adenoma seen to remove surgically, we performed a trial of cinacalcet with successful reduction in PTH and normalization of his calcium and phosphorus levels. Conclusion: Pseudohypoparathyroidism and hypocalcemia are well reported in acrodysostosis. To the best of our knowledge, this is the first reported case of hypercalcemia caused by hyperparathyroidism in a patient with acrodysostosis.
, Amit Akirov, Ilan Shimon
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1298-1303; https://doi.org/10.4158/ep-2020-0171

The publisher has not yet granted permission to display this abstract.
, Adele Amine, Demetrios Herodotou
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0474

Abstract:
Objective: Giant parathyroid adenomas (GPAs) are a rare type of parathyroid adenoma (PA) weighing >2 to 3 g. The objective of this manuscript is to report a case of giant parathyroid adenoma and highlight the risk of postoperative hypocalcemia. Methods: We describe the presentation and work-up of a woman with GPA confirmed with histopathology as well as management of postoperative hypocalcemia. Results: A 66-year-old Caucasian female with chronic fatigue was found to have elevated calcium levels at 13.7 mg/dL (normal, 8.9 to 10.2 mg/dL) and elevated parathyroid hormone levels at 1,240 pg/mL (normal, 12 to 72 pg/mL). Contrast enhanced computed tomography scan of the neck showed a 5.6 cm heterogeneous mass posterior to the right thyroid. The patient underwent right parathyroidectomy and histopathology confirmed the diagnosis of a giant parathyroid adenoma (GPA) weighing 28.7 g. Postoperatively, the patient developed hypocalcemia secondary to hungry bone syndrome and was treated aggressively with intravenous calcium supplementation. At 4 weeks postoperatively, she had normal calcium levels without any calcium supplementation. Conclusion: GPAs are a rare variant of PA, and could be a distinct clinical entity with features different from both PA and parathyroid carcinoma. We further postulate that the risk of postoperative hypocalcemia secondary to hungry bone syndrome could be higher among patients with GPA compared to patients with PA. These hypotheses needs to be validated with further studies.
Stephanie J. Farah, Nohad Masri, Hady Ghanem,
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0452

Abstract:
Objective: Alpelisib-induced DKA is a rare, but life-threatening adverse event. There have been only two reported cases in the literature. We describe such a case, with emphasis on the importance of screening and achieving adequate glycemic control prior to and after initiation of therapy. Methods: A 49-year-old woman, known to have advanced breast cancer, presented with a 3-day history of nausea, vomiting and diffuse abdominal pain. She started alpelisib 300 mg/d two months prior to presentation, after failing other options. She was diagnosed with DKA using her clinical and laboratory features, leading to treatment with hydration and IV insulin therapy. Results: Laboratory data showed high anion gap metabolic acidosis, hyperglycemia and ketonemia with negative GAD-65 antibodies, leading to the diagnosis of alpelisib-associated DKA. Alpelisib was held, and she was treated with IV insulin and hydration. When DKA and hyperglycemia resolved, alpelisib was resumed at a lower dose (200 mg/d) and her blood glucose (BG) managed using a regimen combining insulin and metformin. Conclusion: PI3K signaling is important for the metabolic actions of insulin and alpelisib has been associated with severe hyperglycemia. Metformin is the first line treatment, however when DKA is the presenting syndrome, insulin needs to be considered. BG and HbA1c should be checked prior to treatment initiation and monitored closely after drug initiation. DKA, albeit rare, must be considered in an acutely ill, alpelisib-treated patients presenting with metabolic acidosis, and if drug discontinuation is not an option, insulin treatment may be required to control glycemia.
Sobia Faisal,
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0282

Abstract:
INTRODUCTION: Vitamin D deficiency is quite prevalent worldwide and is usually treated with oral supplementation. Bioavailability of Vitamin D may differ among individuals due to variable absorption and metabolism in the body. CASE PRESENTATION: 66-year-old woman presented for evaluation of low 25(OH)D level. She had no known prior history of gastric/intestinal surgeries or malabsorptive conditions. She had previously been treated with vitamin D3, 2000 IU by mouth (PO) daily with poor response and then vitamin D2 50,000 weekly PO, with persistently low 25(OH)D level at 14 ng/ml after 8 weeks of treatment. Celiac screen was negative and duodenal biopsy was normal. Due to demonstration of poor oral absorption, she was prescribed vitamin D2 50,000 units sublingually for 8 weeks and then changed to over the counter vitamin D3 drops sublingually (1000 units/drop) at 4000 unit twice daily due to suboptimal response with vitamin D2. 25(OH)D level improved gradually to 28 ng/ml after 12 weeks on this regimen and was at 37 ng/ml at 1 year. CONCLUSION: Sublingual vitamin D3 may be an effective alternative mode of vitamin D supplementation in patients who demonstrate poor oral vitamin D absorption despite adequate supplementation for various reasons.
Danielle Logiurato, Zoltan Antal, Ping Zhou
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0354

Abstract:
Objective: Treatment of metastatic adrenocortical carcinoma is challenging and long-term survival rates are exceedingly low. Long-term outcome data for pediatric patients who received mitotane is very limited. Methods: We describe the case of a two-year-old boy with adrenocortical carcinoma with a lung metastasis. He was treated with surgery, chemotherapy and mitotane, and remains disease-free 13 years after diagnosis. Results: Key endocrine issues learned from this case: Adrenal-derived sex-steroid and IGF-2 levels are correlated with disease status; very high doses of glucocorticoid and mineralocorticoid are required while on treatment of mitotane; and central precocious puberty needs to be detected and treated in a timely manner to preserve final adult height. Conclusion: We report a case of pediatric adrenocortical carcinoma with metastasis that was successfully treated with surgery, chemotherapy and adjuvant therapy with mitotane. Appropriate endocrine testing and management are important for long-term survival and quality of life.
, Liana Tripto-Shkolnik
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0234

Abstract:
Objective: Seizures following administration of potent bisphosphonates have been reported only sporadically in medical literature. The rare cases described were often attributed to other precipitating factors such as hypoglycemia, acute infection or predisposition to post-bisphosphonate hypocalcemia. We review the previous cases and present a new case of suspected seizure episode following Zoledronic Acid therapy. Methods: We describe a case of a 63-year-old woman with a history of well controlled epileptic disorder with no seizure activity in recent years. She was treated with intravenous Zoledronic Acid due to osteoporosis. Twelve hours after treatment, she suffered an episode of loss of consciousness with urinary incontinence suspected to be seizure-related. Results: Unlike previously reported cases, our patient had a low risk for post-infusion hypocalcemia as her creatinine, calcium, parathyroid hormone and Vitamin D were all within normal limits prior to the infusion. Conclusions: Our interpretation of the scenario described is based on clinical judgment and not supported by ancillary studies. Nevertheless, our case, along with the limitations described, joins other reports, and raises questions about possible interaction between a convulsion disorder and a potent bone resorption inhibition administration, leading to a relative hypocalcemia and possible seizure threshold reduction. This question should be further explored by other studies.
Feng Xu, Dexing Dai, Ruoman Sun, Zhenming Liu, Xiaolin Lin, Lusha Li, Xiaoping Xing, Xiangbing Wang, ,
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1244-1254; https://doi.org/10.4158/ep-2020-0124

The publisher has not yet granted permission to display this abstract.
Arpita Bhalodkar, Halis Sonmez, Martin Lesser, TungMing Leung, Karina Ziskovich, Damian Inlall, Renee Murray-Bachmann, Marina Krymskaya,
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1331-1336; https://doi.org/10.4158/ep-2020-0261

The publisher has not yet granted permission to display this abstract.
Jeremy D. Purser, Ruba Riachy, Lucas S. Blanton, L. Maria Belalcazar
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0375

Abstract:
Objective: Topical steroid use is common, but its association with Cushing’s syndrome is rare. We report the rapid development of iatrogenic Cushing’s syndrome in a patient on ritonavir who applied a moderate-potency topical steroid cream, triamcinolone, on his genital mucosa for treatment of phimosis. Methods: Clinical and diagnostic challenges associated with topical steroid use are presented and discussed. Results: A 41-year-old man with human immunodeficiency virus infection on stable antiretroviral therapy that included ritonavir, a cytochrome P450 3A4 inhibitor, presented with new onset diabetes and development of overt cushingnoid features over a four-week period. He reported no known history of steroid use. A midnight salivary cortisol using a quantitative enzyme immunoassay was obtained and reported at >15.0 μg/dL (normal <0.112 μg/dL). However, free cortisol in a 24-hour urine collection was undetectable by high-performance liquid chromatography and morning plasma cortisol was also unexpectedly low at 1.1 μg/dL (reference: 4.5 - 23.0 μg/dL). Further investigation revealed that the patient had been applying a topical cream with triamcinolone acetonide (0.1%) on the glans penis for treatment of phimosis. The salivary enzyme immunoassay for cortisol appears to have detected the absorbed triamcinolone, a compound known to cross-react with cortisol in this assay. Conclusion: This case raises awareness on the severe metabolic consequence resulting from the seemingly benign use of a topical steroid medication when applied to the genital mucosa in the setting of stable therapy with ritonavir and illustrates the limitations of salivary cortisol enzyme immunoassays for the evaluation of Cushing’s syndrome in this setting.
, Sunao Matsubayashi, Takeshi Hara, Shinya Satoh, Hiroyuki Yamashita
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0311

Abstract:
Objective: The objective of this report was to describe an unusual case of emerging primary hyperparathyroidism (PHPT) accompanied by recovery of parathyroid blood flow 3 months after spontaneous parathyroid hemorrhage. Methods: Neck images and laboratory tests including serum calcium and parathyroid hormone (PTH) were performed to evaluate parathyroid hemorrhage. Pathologic findings after parathyroidectomy are also presented. Results: A 58-year-old woman developed acute onset of neck pain and swelling with ecchymosis. Computed tomography showed a right para-tracheal hematoma-like lesion behind the thyroid. Ultrasound (US) of the neck revealed a round, hypoechoic nodule measuring 27 × 25 × 18 mm in the right lower thyroid pole without vascular flow. Blood tests showed corrected calcium 9.3 mg/dL (reference range: 8.7–10.3 mg/dL), intact PTH 68 pg/mL (reference range: 10–65 pg/mL). Intact PTH measurement in fine-needle aspirate of the lesion was 339 pg/mL, confirming parathyroid origin. Repeat US after 3 months showed remarkable decrease in lesion size with significant blood flow. Blood biochemistry showed was corrected calcium 10.9 mg/dL, intact PTH 237 pg/mL. She eventually underwent parathyroidectomy, and pathological examination revealed parathyroid adenoma with a tiny thrombus. Conclusion: Spontaneous remission of PHPT after parathyroid hemorrhage has been known to occur sporadically, a phenomenon referred to as autoparathyroidectomy. Although spontaneous remission with permanent improvement of PHPT may be observed, PHPT can recur in the relative short term after parathyroid hemorrhage, and so follow-up blood biochemistry surveillance is requisite. Aso, evaluating parathyroid blood flow using color Doppler US might be useful in verifying the recurrence of PHPT.
, Elchanan Dreyfuss, Rosane Ness-Abramof, Rena Pollack, Avivit Cahn
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1304-1311; https://doi.org/10.4158/ep-2020-0260

The publisher has not yet granted permission to display this abstract.
, Irina Bancos, Amir Hamrahian, Mouhammed Amir Habra, Anand Vaidya, Alice C. Levine,
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1366-1383; https://doi.org/10.4158/dscr-2020-0567

The publisher has not yet granted permission to display this abstract.
Zachary W. Bloomer, Elizabeth M. Bauer, , Mohamed K.M. Shakir
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0225

Abstract:
Objective: Pheochromocytoma is diagnosed biochemically by demonstrating excessive production of catecholamines and their metabolites in the blood and urine. However, these tests are at times fraught with false-positive results due to drug effects. We report here a patient with markedly elevated urinary metanephrines associated with the use of eletriptan for migraine treatment. Methods: A literature search was conducted using the PubMed and Google Scholar databases for eletriptan and false positive metanephrine elevation. Urine and plasma metanephrine tests were performed via liquid chromatography/tandem mass-spectrometry. Results: A 29-year-old man with migraine recently started on eletriptan was evaluated for worsening headache. Initially his blood pressure was 220/160 with a creatinine of 1.9 mg/dL. He was treated with intravenous nicardipine. His lab tests showed normal aldosterone/plasma renin activity ratio, midnight salivary cortisol, thyroid function and urinary drug screen. A 24-hour urine metanephrine 2494 mcg (ref 45–290) and normetanephrine levels 1341 mcg (ref 82–500) for secondary hypertension work up were markedly elevated. In contrast, plasma metanephrines 27 pg/mL (ref 0–62) and normetanephrines 255 pg/mL (ref 0–145) were only mildly elevated. Adrenal CT and Gallium-68 PET/CT showed no abnormalities. Within one week of eletriptan discontinuation, his urine and plasma metanephrine and normetanephrine levels completely normalized as well as reduction of blood pressure (130s/80s mmHg). Conclusion: The discrepancy between plasma and urine studies in our patient suggests the possibility of false positive tests. It is possible that eletriptan may affect the urine assays, but the exact mechanism causing elevated urine metanephrines/normetanephrines is not clear.
Hanting Liang, Hui Miao, Hui Pan, Hongbo Yang, Fengying Gong, Lian Duan, Shi Chen, Linjie Wang, Huijuan Zhu
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1255-1268; https://doi.org/10.4158/ep-2019-0518

The publisher has not yet granted permission to display this abstract.
, Ruth McManus
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0184

Abstract:
Objective: Hirsutism and hyperandrogenism in premenopausal women are most often associated with polycystic ovarian syndrome (PCOS). We present a case of progressive, severe hyperandrogenism with negative imaging identified on surgical histopathology as being due to a Leydig cell tumor, thus illustrating localization challenges associated with these small tumors. Methods: Laboratory investigations included testosterone, dehydroepiandrosterone sulfate (DHEAS), 17α-hydroxyprogesterone, luteinizing hormone (LH), follicle-stimulating hormone (FSH), thyroid stimulating hormone (TSH), 24-hour urine cortisol (UFC), and prolactin. Imaging included pelvic ultrasound, adrenal magnetic resonance imaging (MRI), and computed tomography (CT). Ovarian vein sampling was not available. Results: A 42-year-old woman presented with frontal alopecia, voice deepening, coarse facial hair, and amenorrhea on a background of lifelong oligomenorrhea. Peak testosterone was 30.2 nmol/L (female range < 2.0) with normal DHEAS, 17-OH progesterone, prolactin, UFC and TSH. Transvaginal ultrasound, adrenal MRI and CT thorax/abdomen revealed no androgen source. Testosterone failed to suppress with GnRH agonist. Although no abnormality was seen during oophorectomy, surgical pathology documented a 1.8 cm well-circumscribed hilar Leydig cell tumor. Post-operative testosterone was < 0.5 nmol/L. Conclusion: Although this patient had testosterone levels well into the masculine range, multiple imaging results were negative with a Leydig cell tumor found only after oophorectomy. Leydig cell tumors are rare ovarian stromal tumors and while 50–70% of these tumors produce androgen, size and clinical severity may not be well correlated. This case report illustrates that despite an association with substantially elevated androgen levels, the small size of Leydig cell tumors can result in localization challenges.
Palak Choksi, Brittany L. Gay, David Reyes-Gastelum, Megan R. Haymart, Maria Papaleontiou
Published: 1 November 2020
Endocrine Practice, Volume 26, pp 1237-1243; https://doi.org/10.4158/ep-2020-0123

The publisher has not yet granted permission to display this abstract.
, Sema Hepsen, Bekir Ucan, Güleser Saylam, Erman Cakal
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0113

Abstract:
Objective: Hypercalcemic associated with the use of sodium glucose transporter-2 (SGLT-2) inhibitor is very rare. Only two cases have been reported in the current literature. In these cases hypercalcemia has occurred with the use of SGLT-2 inhibitors taken with thiazide and excessive calcium predisposing it. The objective of this report is to present a case of hypercalcemia and primary hyperparathyroidism, diagnosed after dapagliflozin treatment. Methods: We describe personal history, laboratory parameters, parathyroid USG, 4D CT-MRI and histopathological findings of the patient. Results: A 49-year-old man with 5-year history of type 2 diabetes mellitus (DM), was found to have hypercalcemia with corrected calcium of 11.28 mg/dl [reference range (RR) 8.8–10.6], 6 months after starting dapagliflozin. Previous records showed normocalcemia for many years. Parathormone level was 70.8 pg/ml (RR, 15–65) and 24-hour urinary calcium excretion level was 492 mg/day (RR, 100–300). On parathyroid USG, a 4×9×14 mm hypoechoic lesion inferior to the right thyroid lobe was detected. A 15×10×9 mm oval lesion was observed in the right paratracheal area and at T1 vertebra level on 4D CT-MRI. The patient underwent lower right parathyroidectomy. Histopathological findings confirmed the diagnosis of parathyroid adenoma. Patient was asymptomatic 3 months post-operatively with a normal corrected calcium level of 9.2 mg/dl. Conclusions: SGLT-2 inhibitors could have unmasked the underlying mild hyperparathyroidism. They can increase predisposition to hypercalcemia when used with medications causing it. Volume depletion caused by SGLT-2 inhibitors may also contribute to hypercalcemia. For this reason, calcium levels should be monitored in patients taking SGLT-2 inhibitors.
Tasma Harindhanavudhi, Takashi Takahashi, Anna Petryk, David W. Polly
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0222

Abstract:
Introduction: Dystrophic scoliosis is a serious skeletal manifestation of neurofibromatosis 1 (NF1). The condition requires surgical intervention that is frequently associated with poor outcome due to the high rate of impaired bone healing, pseudoarthrosis, and loosening of the spinal instrumentation. New therapeutic approaches are needed to improve surgical outcomes. Methods: Clinical, laboratory and radiographic data are presented. Results: A 54-year-old woman with severe NF1 related dystrophic scoliosis and three prior surgical interventions underwent revision of lumbar fusion with intraoperative rhBMP-2 for loosening and a fracture of the left vertical rod at the L4 pedicle screw connection. Two days after surgery, CT scan revealed left posterior iliac periscrew fracture. Given a high risk of mechanical failure, zoledronic acid and asfotase alfa were also administered at 3 and 7 months after surgery. At 14 months after surgery back pain improved, and a CT scan showed stable spinal fusion and healed left posterior iliac screw fracture. Conclusion: Combination therapy including asfotase alfa with rhBMP-2 and bisphosphonate resulted in solid arthrodesis after spinal surgery in NF1 related dystrophic scoliosis.
, Alanoud Alanazi, Zahra A. Al Saeed, Fatimah M. Alabdrabalnabi
Published: 1 November 2020
AACE Clinical Case Reports, Volume 6; https://doi.org/10.4158/accr-2020-0141

Abstract:
Objective We are reporting a case of adipsic diabetes insipidus (ADI) post astrocytoma resection. Method Clinical and laboratory data are presented Results 16-year-old female with a history of incompletely resected hypothalamic astrocytoma admitted with headache. Head MRI showed interval increase in suprasellar lesion with extension to the third ventricle. Following second stage resection, she developed increased urine output with diluted urine resulting in negative fluid balance, however, she was unable to sense thirst. Blood tests showed serum sodium of 155 mEq/dl (136–145), serum osmolality at 321 mOs/kg ( 285–295 ) and urine osmolality of 128 mOsm/kg (300–1600 ). Serum creatinine and potassium were normal. Pituitary hormone profile were normal : Growth Hormone 0.171 ng/ml (0.123–8.05), LH 3.44 miu/ml (7.59–89.08), FSH 5.60 miu/ml (2.55–16.69) TSH 2.9 miu/ml (0.35–4.94), Free Thyroxine 0.92 ng/dl (0.7–1.48), ACTH 19.56 pg/ml (7.2–63.3) and Prolactin 7.25 ng/ml (5.18–26.53). The patient was treated with Desmopressin Acetate 120 mcg tablets twice daily with a fixed fluid intake of 1.5 to 2.0 liters/day with closed monitoring of fluid intake, output and body weight. Response was good with gradual reduction of serum sodium level of around 7–9 mEq/L/Day. Conclusions Adipsic diabetes insipidus is a rare entity of central diabetes insipidus, where the absence of polydipsia can be challenging in diagnosing and managing the condition. Cases of ADI are likely under reported and clinicians need to be aware of this condition
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