Case Reports in Endocrinology最新文献

Overdose of long-acting insulin can cause unpredictable hypoglycemia for prolonged periods of time. The initial treatment of hypoglycemia includes oral carbohydrate intake as able and/or parenteral dextrose infusion. Refractory hypoglycemia following these interventions presents a clinical challenge in the absence of clear guidelines for management. Octreotide has sometimes been used, but its use is generally limited to sulfonylurea overdose. In this case report, we present a case of refractory hypoglycemia following an overdose of 900 units of long-acting insulin glargine that failed to respond to usual modes of therapy mentioned above. Stress-dose corticosteroids were then initiated, followed by subsequent improvement in IV dextrose and glucagon requirements and blood glucose levels. Hence, corticosteroids may serve as an adjunctive therapy in managing hypoglycemia and can be considered earlier in the course of treatment in patients with refractory hypoglycemia to prevent volume overload, especially when large volumes of dextrose infusions are required.

The use of dipeptidyl peptidase-4 inhibitors (DPP4i) appears to be associated with a small but significantly elevated risk of bullous pemphigoid (BP). Although the pathogenic mechanism of DPP4i-associated BP remains unclear, this adverse event is reported with multiple gliptins, suggesting a class effect. However, previous studies from various countries showed that vildagliptin had been implicated in most cases. The aim of this study was to illustrate a case series of DPP4i-associated BP in Thai patients. We conducted a retrospective study from consecutive cases of BP in people with type 2 diabetes mellitus (T2DM) from January 2008, the year in which the first DPP4i was introduced in Thailand, until December 2019. During the study period, 10 BP patients with T2DM were identified. A total of 5 DPP4i-associated BP (3 on vildagliptin, 1 on linagliptin, and 1 on sitagliptin) were found. All patients were male with a mean age at BP development of 80.4 years (73-86 years). All patients had a long-standing duration of diabetes (median duration 34 years), and mean A1C was 7.5 ± 1.4%. The median time to BP development after the introduction of DPP4i was 64 months (range 20-128 months). The severity of BP was classified as mild in 2 cases. In all cases, the association between the drug intake and BP onset was classified as "possible" according to the Naranjo causality scale. All of the patients continued taking DPP4i after BP diagnosis, and one patient died of lung cancer 18 months after BP diagnosis. Only 2 patients could achieve complete remission at least 2 months after stopping DPP4i. Our case series demonstrated a potential link between DPP4i and the development of BP, which mainly occurred in very elderly male patients. The latency period from an introduction of DPP-4i could be several years, and the clinical course after DPP4i discontinuation varied. Clinicians prescribing DPP4i should be aware of this association and consider stopping this medication before a refractory disease course ensues.

Over 50% of patients with papillary thyroid carcinoma (PTC) have cervical lymph-node metastasis on diagnosis, and up to 30% show nodal recurrence after surgery plus radioactive iodine (131I) (RAI) therapy. The combination of ultrasonography (US) and fine-needle aspiration cytology (FNAC) and the measurement of thyroglobulin (Tg) in washout fluid are cornerstones in the diagnosis of nodal metastasis. In the absence of anti-Tg antibodies, unstimulated serum thyroglobulin (Tg) levels are generally a reliable marker of recurrent disease, and 18F-FDG positron emission tomography (PET)/computed tomography (CT) plays an important role in the imaging work-up. We report the case of a 65-year-old man evaluated for a large multinodular goitre which caused compressive symptoms; the dominant nodule in the left lobe presented suspicious features on US. Thyroid function showed subclinical hypothyroidism, calcitonin was normal, serum thyroglobulin levels were low, and anti-thyroid antibodies were absent. The prevalent left nodule showed an intense uptake on 18F-FDG PET/CT but proved benign at FNAC. On the basis of the suspicious clinical and imaging features, total thyroidectomy was performed. Histology revealed a tall-cell variant of PTC with scattered expression of Tg and diffuse high expression of cytokeratin (CK) 19; RAI therapy was performed. Within 6 years of surgery, left laterocervical lymph-node recurrence was twice detected (first at levels II and III, then at levels IV and VI) by US and 18F-FDG-PET/CT and was confirmed by FNAC. Tg levels in the washout fluid proved clearly diagnostic of metastasis only in the second, larger, recurrence, whereas serum Tg levels (in the absence of anti-Tg antibodies) always remained undetectable on L-thyroxine therapy. Surgery was performed on both recurrences, and histology confirmed lymph-node metastasis of PTC. Immunohistochemical expression of Tg and CK 19 was similar to that of the primary tumour. No further relapses have occurred to date. Posttherapy (surgery and RAI) unstimulated serum Tg levels may not be a reliable marker of nodal recurrence in patients with differentiated thyroid cancer (DTC) that produces low amounts of Tg.

Familial hypocalciuric hypercalcemia (FHH) is considered a relatively benign condition characterized by mild elevations in serum calcium and relatively low urinary calcium excretion. It results from an elevated set point in serum calcium arising from variants in the calcium-sensing receptor (CaSR) gene but also AP2S1 and GNA11 genes, which encode for adaptor-related protein complex 2 and G11 proteins, respectively. The manifestations of FHH can vary and sometimes overlap with primary hyperparathyroidism making the diagnosis challenging. Case Presentations. We report a mother and daughter with a novel heterozygous variant in the CaSR gene resulting in a serine to leucine substitution at position 147 (S147L) of the CaSR. Both patients had mild hypercalcemia, relatively low urinary calcium excretion, elevated calcitriol, and low-to-normal intact PTH. The proband (daughter) presented with symptoms associated with hypercalcemia and was incidentally found to have a bony lesion suspicious for osteitis fibrosa cystica, and she was also diagnosed with sarcoidosis. Subtotal parathyroidectomy revealed normal-weight parathyroid glands comprised of 50-80% parathyroid epithelial cells, which has been documented as within the spectrum of normal. Her mother had no symptoms, and no intervention was pursued. Conclusion. We report a novel variant in the CaSR associated with FHH in two patients with similar biochemical features yet differing clinical manifestations. While the relationship of the bony findings and parathyroid histology with this variant remains unclear, these cases enrich our knowledge of CaSR physiology and provide further examples of how varied the manifestations of FHH can be.

Subacute thyroiditis is a self-limiting inflammatory disorder, characterized by neck pain or discomfort, a tender diffuse goiter, and sometimes a transient episode of hyperthyroidism followed by euthyroidism and sometimes hypothyroidism. There is usually a normalization of thyroid function within a few weeks. Subacute thyroiditis has a higher incidence in summer and has been linked to a viral or bacterial upper respiratory postinfection inflammatory response. We hereby describe the case of a previously healthy 37-year-old female presenting with subacute thyroiditis associated with a very mild presentation of COVID-19. As most patients with SARS-Cov-2 are asymptomatic, we suggest to rule out SARS-Cov-2 infection in patients presenting with symptoms suggesting SAT.

Adrenocortical oncocytomas are rare and mostly nonfunctioning neoplasms. We report the case of a 27-year-old woman diagnosed with an ACTH-independent Cushing's syndrome due to left adrenal oncocytoma. She underwent laparoscopic adrenalectomy. Histopathological examination revealed an oncocytoma of uncertain malignant potential with a low Ki-67 proliferation index, inhibin A positivity, and chromogranin A negativity. Electron micrographs confirmed adrenal oncocytoma cells, characterized by the presence of a large amount of mitochondria. The postoperative course was uneventful, and the patient experienced a progressive regression of Cushing-related symptoms. Periodical follow-ups with MRI and cortisol dosage are required due to the neoplasm's uncertain malignant potential. Considerations on the diagnosis, pathology findings, clinical remarks, and interventions are made.

Pheochromocytomas are tumors that originate from the chromaffin tissue of the adrenal medulla and commonly produce catecholamines. The diagnosis is typically established by the measurement of catecholamines or their metabolites in urine or plasma, and tumors are localized with the use of radiographic and scintigraphic studies. Pheochromocytomas can occur in asymptomatic patients, and the preferred treatment is surgical removal of the tumor. We report a 48-year-old male with a left adrenal incidentaloma, which progressively increased in size from 1.1 cm to 2.6 cm over a 4-year period, as measured by an adrenal computed tomography (CT) scan. Throughout his entire course of treatment, he was asymptomatic with normal blood pressure readings. His biochemical screening was unremarkable for the first three years of tumor surveillance. Follow-up imaging, including CT and MRI, showed findings suspicious for pheochromocytoma, and the diagnosis was ultimately made with the combination of imaging and laboratory studies. He underwent laparoscopic resection of the adrenal mass with confirmation of pheochromocytoma on histology. This case illustrates how CT and MRI findings can alert providers to the presence of a pheochromocytoma, even in an asymptomatic, biochemically negative patient.

A 38-year-old gentleman presented with thyroid storm with multiorgan involvement in the form of heart failure (thyrotoxic cardiomyopathy), respiratory failure (respiratory muscle fatigue), hepatic dysfunction, fast atrial fibrillation, pulmonary embolism, and disseminated intravascular coagulation (DIC). His Graves' disease (GD) remained undiagnosed for nearly 8 months because apart from weight loss, he has not had any other symptoms of thyrotoxicosis. The presentation of thyroid storm was atypical (apathetic thyroid storm) with features of depression and extreme lethargy without any fever, anxiety, agitation, or seizure. There were no identifiable triggers for the thyroid storm. Apart from mechanical ventilation and continuous veno-venous renal replacement therapy in the intensive care unit, he received propylthiouracil (PTU), esmolol, and corticosteroids, which were later switched to carbimazole and propranolol with steroids being tapered down. He was diagnosed with thyrotoxic myopathy which, like GD, remained undiagnosed for long (fatigability). A high index of suspicion and a multidisciplinary care are essential for good outcome in these patients.

Introduction: Hypoparathyroidism, which is a common complication following total thyroidectomy can be transient in majority and permanent in 1.5% of the patients and usually occurs secondary to an inadvertent removal of parathyroid glands, mechanical or thermal injury or disruption of the vasculature. In some patients, it is observed that symptoms of hypocalcemia can occur for the first time several years after the surgery, which is known as "delayed hypoparathyroidism." We report three cases of delayed hypoparathyroidism from Sri Lanka, presenting several years after total thyroidectomy. Case Presentation. Case 1- a 60-year-old Sri Lankan woman who presented with symptomatic hypocalcemia for the first time, 30 years after total thyroidectomy for follicular thyroid carcinoma. Case 2- a 53-year-old Sri Lankan woman presenting with neuropsychiatric manifestations of hypocalcemia for the first time, 12 years after total thyroidectomy for papillary thyroid carcinoma. Case 3- a 49-year-old Sri Lankan woman developing symptoms of hypocalcemia for the first time, 11 years after completion of thyroidectomy for papillary thyroid carcinoma. All these patients were detected to have low parathyroid hormone levels, without an alternative etiology for hypoparathyroidism, hence leading to a diagnosis of delayed post-thyroidectomy hypoparathyroidism.

Conclusion: Delayed hypoparathyroidism is a rare phenomenon, which is secondary to progressive atrophy of parathyroid glands and slowly progressive hypovascularization of parathyroids due to scar tissue retraction following thyroidectomy. The nonspecific nature of hypocalcemic symptoms and lack of continuous follow-up for a long time after thyroidectomy could contribute to a further delay in diagnosis. However, it is an important diagnosis to consider in any patient with a history of neck surgery presenting with hypocalcemia, irrespective of the time duration of surgery, as timely diagnosis and treatment can prevent long-term complications of hypocalcemia and improve the quality of life.

[This corrects the article DOI: 10.1155/2020/4768281.].