This case report describes a 55-year-old woman presenting with acute dysphagia and neck swelling after a “popping” sensation in her neck. CT and MRI revealed a large retropharyngeal cystic mass and a left thyroid lobe mass, causing airway displacement. Despite significant effacement of the oropharyngeal and supraglottic airway on flexible nasopharyngolaryngoscopy, the patient remained asymptomatic regarding respiratory distress. Subsequent ultrasound-guided fine needle aspiration biopsy diagnosed malignant papillary thyroid carcinoma. Concurrently, previously undiagnosed Graves’ disease was identified. The clinical presentation was attributed to suspected rupture or hemorrhage of the malignant thyroid tumor, leading to a serosanguineous fluid collection and inflammatory edema. The patient was managed with high-dose steroids and intravenous antibiotics, resulting in improved airway edema. She never developed stridor or dyspnea throughout her hospital course and later underwent a total thyroidectomy with neck dissection. This case highlights the potential for a thyroid malignancy to present with suspected spontaneous rupture or hemorrhage causing airway effacement and raises the question of whether Graves’ disease, due to increased vascularity, put patients at additional risk of this presentation.
Introduction Insulin autoimmune syndrome (IAS, Hirata’s disease) is a rare cause of endogenous hyperinsulinemic hypoglycemia classically linked to high-titer insulin autoantibodies and exposure to sulfhydryl (–SH)–containing drugs. IAS is strongly HLA-associated and is most frequently reported in East Asian populations. We report a case in which simultaneous exposure to two thiol-containing agents unmasked IAS in a non-Asian patient carrying HLA-DRB1*04:06. Case presentation A 60-year-old woman developed recurrent autonomic and neuroglycopenic episodes 2–4 hours after meals. Home medications included captopril and over-the-counter α-lipoic acid (ALA). During a symptomatic episode her plasma glucose was 38 mg/dL with markedly elevated serum insulin, C-peptide and proinsulin; mixed-meal testing reproduced a postprandial glucose peak followed by profound hypoglycemia accompanied by paradoxical hyperinsulinemia. Insulin autoantibodies were strongly positive (>150 U/mL) and HLA genotyping revealed DRB1*04:06. A supervised fast was non-diagnostic for insulinoma. Management comprised immediate discontinuation of both captopril and ALA (captopril replaced with losartan) and dietary measures (frequent small low-carbohydrate meals). Hypoglycemic episodes abated within two weeks and resolved by six weeks; fasting glucose, insulin and IAA titers had substantially improved by three months. Conclusion To our knowledge, this is the first reported case of IAS temporally associated with concurrent captopril and ALA exposure in a non-Asian carrier of HLA-DRB1*04:06. The case highlights the need to review both prescription drugs and supplements when investigating hyperinsulinemic hypoglycemia, that IAS can occur outside typical ethnic groups when permissive HLA alleles are present, and that prompt withdrawal of offending agents with conservative measures often leads to spontaneous remission.
Introduction Paraneoplastic pemphigus (PNP) is an uncommon autoimmune blistering disorder frequently associated with underlying malignancies. Renal cell carcinoma (RCC) is the most common primary renal cancer. It is associated with a wide range of paraneoplastic syndromes, but occasionally presents as a dermatological finding. Case presentation A 56-year-old woman with severe oral and cutaneous lesions was diagnosed with PNP associated with renal cell carcinoma (RCC). The patient was treated with 1 g of rituximab as an infusion, and after two weeks, another dose of 1 g of rituximab was administered in combination with steroids (prednisolone 0.5 mg/kg) until control, then gradually decreasing by 25% each month, until we reached 5 mg/day. The patient achieved complete clinical remission at 6 months; however, long-term outcomes remain unknown because she was subsequently lost to follow-up. Conclusion A comprehensive workup is crucial for diagnosing the underlying tumor associated with this phenomenon. Rituximab- and steroid-based therapies may be effective in controlling PNP symptoms.
Introduction Thyrotoxic periodic paralysis (TPP) is an uncommon endocrine emergency caused by rapid intracellular potassium redistribution during thyrotoxicosis. Carbohydrate exposure is a recognized precipitant, but direct observation of a first paralytic attack developing during supervised diagnostic glucose testing is unusual. Case Presentation A 29-year-old Indian man underwent a supervised oral glucose tolerance test (OGTT) for borderline fasting dysglycemia and episodic postprandial tremulousness. He had also experienced recent weight loss, heat intolerance, and palpitations but had no previous weakness. During the late phase of testing, progressive thigh heaviness evolved into symmetrical flaccid quadriparesis with lower-limb predominance, reduced reflexes, preserved sensation, and no respiratory involvement. Serial samples demonstrated a rapid transition from normal baseline potassium to severe hypokalemia, accompanied by hypophosphatemia and a marked insulin response. Normal acid-base status and suppressed urinary potassium excretion supported transcellular redistribution. Thyroid testing and antibody studies established previously undiagnosed Graves’ disease. Limited potassium replacement, magnesium supplementation, and propranolol produced complete neurological recovery without rebound hyperkalemia. Carbimazole therapy achieved progressive biochemical control, with no recurrent paralysis during 24 weeks of follow-up. Discussion Standardized glucose exposure and serial sampling documented the metabolic transition from normal potassium to paralytic hypokalemia. The episode supports an interaction between endogenous insulin stimulation and thyrotoxic skeletal-muscle susceptibility rather than hyperinsulinemia as an isolated cause. Conclusion Unrecognized thyrotoxicosis should be considered before carbohydrate provocation testing in patients with unexplained hyperadrenergic symptoms.
Anesthesia mumps, also known as acute postoperative sialadenitis, is a rare complication that can occur following general or regional anesthesia. The prevalence of this condition ranges from 0.16% to 0.2%. This case report presents a patient with recurrent bilateral parotid swelling after cesarean section (CS) under spinal anesthesia and its management. A 42-year-old G5P4 woman with gestational diabetes mellitus and antiphospholipid syndrome presented for elective cesarean delivery at 39 weeks and two days of gestation under spinal anesthesia. The patient developed bilateral facial swelling after five hours, which was more evident on the right parotid site. A review by an otolaryngologist confirmed the diagnosis of anesthesia mumps. Anesthesia mumps typically presents as painless swelling of the salivary glands. Salivary duct obstruction and hyperemia are believed to be common causes. This case highlights that anesthesia-induced mumps is a reversible and transient complication that may occur after short surgical procedures, such as cesarean sections. Early recovery was achieved following supportive conservative management, with hydration and analgesia.
