Locantore Pietro, Oliva Alessandro, Cera Gianluca, Paragliola Rosa Maria, Novizio Roberto, Policola Caterina, Corsello Andrea, Pontecorvi Alfredo
Unit of Endocrinology, Department of Translational Medicine and Surgery, Università Cattolica del Sacro Cuore, Fondazione Policlinico "A. Gemelli" IRCCS, Largo Gemelli 8, 00168 Rome, Italy.
Unicamillus, Saint Camillus International University of Medical Sciences, Via di S. Alessandro 10, 00131 Rome, Italy.
Diseases. 2025 Jan 17;13(1):20. doi: 10.3390/diseases13010020.
Hypercalcemia is a frequently encountered laboratory finding in endocrinology, warranting accurate clinical and laboratory evaluation to identify its cause. While primary hyperparathyroidism and malignancies represent the most common causes, many other etiologies have been described, including some reports of hypercalcemia secondary to adrenal insufficiency. On the contrary, hypoparathyroidism is a relatively common cause of hypocalcemia, often arising as a complication of thyroid surgery. In real-world clinical practice, however, many challenges come into play, and a comprehensive approach may not be enough to establish a diagnosis. we describe a peculiar case of severe hypercalcemia occurring in a 47-year-old woman with a previous history of post-surgical permanent hypoparathyroidism treated with calcitriol (0.5 µg bid) and calcium carbonate (1 g qd), which persisted after withdrawal of these drugs. During her follow-up, an ACTH-independent Cushing syndrome was diagnosed, leading to a unilateral right adrenalectomy. In the two months following surgery, she was admitted to the emergency ward on three occasions because of severe, persistent, idiopathic hypercalcemia. On each occasion, parathyroid hormone levels were confirmed to be undetectable, with low vitamin D levels. Common and rare causes of hypercalcemia were excluded, and the persistence of severely elevated calcium levels led to the empirical use of intravenous clodronate, achieving remission of both hypercalcemia and, unexpectedly, hypoparathyroidism. After 8 months, due to borderline-reduced calcium, calcitriol at 0.5 µg qd was restarted. After 18 months of follow-up, the patient is well and normocalcemic, with low-dose calcitriol. Notably, the patient had no acute adrenal insufficiency, distinguishing this case from other post-adrenalectomy hypercalcemia reports. the history of hypoparathyroidism makes this case even more unusual, and it encourages careful follow-up of hypoparathyroid patients with Cushing syndrome. Ongoing observation, as well as new research on the physiopathology of cortisol and calcium metabolism, are needed to clarify the pathogenesis of this case.
高钙血症是内分泌学中常见的实验室检查结果，需要进行准确的临床和实验室评估以确定其病因。虽然原发性甲状旁腺功能亢进和恶性肿瘤是最常见的病因，但也描述了许多其他病因，包括一些继发性肾上腺功能不全导致高钙血症的报道。相反，甲状旁腺功能减退是低钙血症相对常见的病因，常作为甲状腺手术的并发症出现。然而，在实际临床实践中，会出现许多挑战，仅采用综合方法可能不足以确诊。我们描述了一例特殊病例，一名47岁女性，既往有手术后永久性甲状旁腺功能减退病史，曾接受骨化三醇（0.5μg，每日两次）和碳酸钙（1g，每日一次）治疗，停用这些药物后高钙血症仍持续存在。在她的随访过程中，诊断出促肾上腺皮质激素非依赖性库欣综合征，随后进行了右侧肾上腺切除术。术后两个月内，她因严重、持续的特发性高钙血症三次入住急诊病房。每次甲状旁腺激素水平均证实无法检测到，维生素D水平较低。排除了高钙血症的常见和罕见病因，严重升高的钙水平持续存在导致经验性使用静脉注射氯膦酸盐，高钙血症和意外出现的甲状旁腺功能减退均得到缓解。8个月后，由于血钙水平临界降低，重新开始使用0.5μg每日一次的骨化三醇。经过18个月的随访，患者情况良好，血钙正常，服用低剂量骨化三醇。值得注意的是，该患者没有急性肾上腺功能不全，这使得该病例与其他肾上腺切除术后高钙血症报告有所不同。甲状旁腺功能减退病史使该病例更加不同寻常，它促使对患有库欣综合征的甲状旁腺功能减退患者进行仔细随访。需要持续观察以及对皮质醇和钙代谢生理病理学的新研究来阐明该病例的发病机制。
