Journal of Clinical and Translational Endocrinology: Case Reports最新文献

Sodium-glucose cotransporter-2 (SGLT2) inhibitors for the treatment of type 2 diabetes mellitus are gaining popularity due to their cardioprotective and renoprotective effects, yet SGLT2 inhibitors are increasingly associated with euglycemic diabetic ketoacidosis, which could be life-threatening if missed due to pseudo-normoglycemia. We herein report a 55-year-old male with poorly controlled type 2 diabetes mellitus on Alogliptin, Pioglitazone, Canagliflozin and Repaglinide presented with 3 weeks of low-grade fever, generalized weakness, anorexia, cough, feeling of discomfort in the jaw and dysphonia. The patient was found to have euglycemic diabetic ketoacidosis complicated by subacute thyroiditis. After saline infusion with IV glucose and insulin, he recovered quickly and was discharged on the 8th day of admission. This is the first reported case of simultaneous development of euglycemic DKA and subacute thyroiditis and highlights the importance of identifying the rare combination of disease as soon as possible and treating them promptly.

Hyperparathyroidism may lead to skeletal (osteitis fibrosa cystica) and renal complications; however, these are now uncommon in developed countries where hypercalcemia is detected earlier on biochemical screening and prior to the development of overt symptoms and signs. Rarely, however, these complications may be the presenting problem for a patient with undiagnosed hyperparathyroidism.

We describe the case of a patient with a right tibial mass, severe hypercalcemia and numerous skeletal lytic lesions which could easily have been mistaken for metastatic bone disease. The presence of an elevated parathyroid hormone and histological assessment led to the diagnosis of a brown tumour due to hyperparathyroidism. All patients with hypercalcemia should undergo a systematic workup to avoid missing a benign, treatable disorder.

Ectopic parathyroid glands are not rare. They are usually located between the mandible and mediastinum, and usually localized with standard imaging modalities (^99mTc-sestamibi subtraction scintigraphy and 4D-CT). We report the case of a 67-year-old woman with severe hypercalcemia due to symptomatic primary hyperparathyroidism whose preoperative ^99mTc-sestamibi and 4D-CT scans were non-localizing. During 4-gland exploration, one hypercellular parathyroid was excised, two normocellular parathyroids were biopsied, and bilateral low internal jugular vein sampling showed similar parathyroid hormone (PTH) levels. Despite treatment with zolendronic acid and cinacalcet, symptomatic PHPT persisted. A^99mTc-sestamibi study using a modified anterior and posterior whole-body scintigraphy protocol with SPECT/CT revealed mild focal uptake in the right para-tracheal area, correlating with a mass on SPECT/CT. Sestamibi uptake was visually more conspicuous on posterior projection whole-body image. Thoracoscopic resection of this mass revealed a parathyroid gland (2.6 × 1.5 × 0.7 cm) composed almost entirely of chief cells. The lack of identifiable oxyphil content could explain the lack of or limited radiotracer uptake. The modified protocol differed from standard parathyroid imaging in the timing of radiotracer administration, timing of image capture, and types of images obtained could account for the eventual visualization of the ectopic tissue, and may be considered in challenging cases of suspected ectopic parathyroid gland.

Background

Pituitary apoplexy is a rare endocrine emergency, which commonly presents with headache and is occasionally associated with visual disturbances. Prompt diagnosis and treatment can be both life and vision saving. In the emergence of novel coronavirus and global pandemic, rapid development of new vaccines have shown to reduce morbidity and mortality associated with Covid-19. Recognition of rare potential adverse effects of these vaccines including pituitary apoplexy are yet to be reported. A causal link between pituitary apoplexy and COVID-19 vaccination has not been established.

Case presentation

We report a case of a 24-year-old woman who presented with progressively worsening headache soon after completing her COVID-19 vaccination. Imaging showed pituitary apoplexy with an underlying pituitary mass. In view of the age and the typical presentation of severe headache, pituitary hypophysitis was considered, despite the absence of the almost pathognomonic feature of a thickened pituitary stalk in the initial imaging. In the context that the headache had started shortly after the administration of the second dose of COVID-19 vaccine, this potentially could have been the trigger for the occurrence of pituitary apoplexy.

Conclusion

Although the pathophysiology is not entirely clear and no direct link could be ascertained, our patient may have developed an exaggerated immunological response after the vaccine, with a possible pituitary hypophysitis leading to a pituitary apoplexy.

Background

Monoclonal antibodies have become a mainstay in treatment of autoimmune and malignant disease. Adverse reactions to antibody therapy are often unpredictable and include endocrinopathies. The monoclonal antibody alemtuzumab, which is licensed for the treatment of MS, induces Graves' disease in 22% of patients with MS receiving the drug. Alemtuzumab targets CD52 which causes depletion of mature lymphocytes. In this case report, clinical considerations and pathophysiology are explored by commentating on a patient who developed Graves' disease following alemtuzumab therapy.

Case description

SK is a 37-year-old woman diagnosed with MS at 25, and also suffers from depression. SK began alemtuzumab treatment at aged 35, subsequently, she was found to have deranged TFTs (TSH <0.01 mU/L and fT4 28 pmol/L). SK experienced regular palpitations, fatigue and disturbed sleep. The patient attributed her fatigue and palpitations to MS and did not seek medical attention. SK reported no other symptoms of hyperthyroidism. Symptoms resolved following administration of carbimazole and propranolol.

Discussion

The exact mechanisms underlying both MS and alemtuzumab-induced Graves' disease are unknown. Lymphocyte depletion and reconstitution is thought to cause autoimmunity by over-repopulation of naïve B-cells, T-lymphocyte modulation and disrupting cytokine signalling (IL-21 and IFNy). The slow regeneration of Treg cells that normally promote tolerance underlies these changes. This report identifies challenges including: identifying adverse reactions to new medications, patients discriminately reporting symptoms and the fragility of quality of life in patients with MS.

A rare complication of thyrotoxicosis among Asians is Thyrotoxic Periodic Paralysis (TPP), with an incidence of approximately 2% in patients with thyrotoxicosis from any cause. TPP is characterized by sudden onset of hypokalemia and muscle paralysis. Hypokalemia in TPP results from an intracellular shift of potassium induced by the thyroid hormone sensitization of Na⁺/K⁺–ATPase rather than depletion of total body potassium. Treatment of TPP includes correcting the underlying hyperthyroid state, prevention of potassium shift by using non-selective beta-blockade, and replacing potassium. TPP is curable once a euthyroid state is achieved. We describe here a rare case of TPP in a young Chinese man who presented with sudden bilateral lower limb weakness.

Diabetes insipidus (DI) is a rare clinical condition in the postoperative period. Post-surgery polyuria is a common finding, as the body excretes the excessive fluid given during surgery. It is important to diagnose and differentiate the DI from post-operative polyuria, as DI can lead to severe dehydration and electrolyte disturbances. We report two unusual cases of perioperative DI requiring desmopressin therapy.

Case 1

A 46-year-old healthy male patient developed intraoperative DI leading to hypernatremia during the anterior cervical discectomy and fusion. Anesthesia was maintained with propofol and remifentanil target-controlled infusion (TCI). After two hours of surgery, the patient became polyuric and was passing diluted urine. He received desmopressin and hydration. The patient recovered and was transferred to the ward, then, discharged home without any clinical or neurological problems.

Case 2

A 36-year-old healthy male patient underwent elective 3rd ventricular cyst excision. Pre-anesthesia assessment did not reveal any comorbidities and the surgery was uneventful. His anesthesia was maintained with propofol and remifentanil TCI (target-controlled infusion). In the postoperative period, he developed DI requiring hydration and desmopressin. The patient's further recovery was uneventful. He was discharged home through the ward.

Conclusion

The occurrence of DI in the above-mentioned surgeries is very rare. Both surgical procedures and anesthesia medications can cause perioperative DI.

Introduction

Although Graves' disease (GD) is the most common cause of hyperthyroidism in adolescents, it is very rare for it to result from the production of thyroid-stimulating hormone (TSH) receptor autoantibodies due to Graves' immune reconstitution inflammatory syndrome (IRIS). Especially for paediatric patients, very little is known about the aetiology and complete pathogenesis of Graves’ IRIS. Furthermore, details of a valid treatment plan are severely lacking. The case report presented here is only the third for paediatric patients worldwide.

Case presentation

We report on a Caucasian female adolescent who initially presented with non-specific complaints about discomfort and tightness in the anterior part of the neck and thyroid enlargement. Based on clinical, laboratory and thyroid ultrasound findings, she was diagnosed with GD. However, after several months of outpatient treatment, the patient's GD could still not be fully managed with conservative therapy alone. Only when the patient was hospitalized for the third time was it discovered that she had previously been diagnosed with human immunodeficiency virus infection and had received highly active antiretroviral therapy (HAART) for the previous 29 months. Consequently, the production of autoantibodies to TSH receptors and abnormal changes in thyroid hormones had led to the development of GD and her final diagnosis was established as Graves' IRIS. Ultimately, a total thyroidectomy was performed.

Discussion/conclusion

This case report demonstrates how fundamentally important it is to have full access to a patient's complete anamnesis and results of all previous investigations. Clinicians should be made aware of the potential existence of thyroid dysfunction and other autoimmune or infectious processes in paediatric patients initiating or reinitiating HAART. Further research is needed to optimize the treatment for such paediatric patients.

Background

Hypothyroidism is a well-known side effect associated with tyrosine kinase inhibitors (TKIs) therapy. We describe a case of a patient with a history of postsurgical hypothyroidism who presented TSH elevation ranging from 1.68 to 17.09 IU/ml with normal free thyroxine (T4) after starting treatment with alectinib for non-small cell lung cancer (NSCLC).

Case presentation

A 78-year-old female, with past medical history of Graves’ disease with subsequent total thyroidectomy and residual postsurgical hypothyroidism, was diagnosed with non-small cell lung cancer (NSCLC) and presented with TSH elevation with normal free T4 after starting therapy with alectinib.

Conclusion

The most likely etiology for TSH elevation during her therapy with alectinib is an increased requirement of thyroid hormone secondary to increase activity of type 3 deiodinase and/or inhibition of monocarboxylate transporter 8 (MCT8) with consequent lower tissue availability of active T3. Further studies of thyroid dysfunction after alectinib administration are lacking.

Doege-Potter syndrome (DPS) is a rare paraneoplastic entity associated with solitary fibrous tumors which are mesenchymal spindle cell neoplasms. DPS is characterized by non-insulin mediated severe hypoglycemia due to release of the prohormone big Insulin like growth factor (IGF)- 2. Here, we present the case of a 59-year-old gentleman who was evaluated for frequent episodes of hypoglycemia. Further work up revealed low insulin, IGF-1 and IGF-2 levels and a large left intrathoracic solitary fibrous tumor. After resection of the mass, his hypoglycemic episodes completely resolved.