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一名糖尿病患者合并功能性吉特曼综合征的常染色体显性遗传性钙蛋白酶病

Autosomal Dominant Calpainopathy in a Diabetic Patient Complicated by Functional Gitelman Syndrome.

作者信息

Peterpalaniswami Kabilash Manivalli, Madhavan Krishnaswamy, Mathew Gerry George, Jayaprakash V

机构信息

Department of General Medicine, SRM Medical College Hospital and Research Centre, Kattankulathur, Kanchipuram 603203, Tamil Nadu, India.

Department of Nephrology, SRM Medical College Hospital and Research Centre, Kattankulathur, Kanchipuram 603203, Tamil Nadu, India.

出版信息

Case Rep Med. 2025 Jun 5;2025:4210190. doi: 10.1155/carm/4210190. eCollection 2025.

DOI:10.1155/carm/4210190
PMID:40520344
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12163201/
Abstract

Adult-onset Gitelman syndrome with calpainopathy is a rare clinical condition in patients with diabetes mellitus. We present the case of a 52-year-old male diabetic patient who presented with muscle weakness and fatigue. On evaluation, he had reduced power in the thigh and pelvic girdle muscles. Laboratory tests revealed hypokalemia, hypomagnesemia, metabolic alkalosis, kaliuresis, and hypocalciuria, which led to the diagnosis of Gitelman syndrome. Electromyography revealed a myopathic pattern with polyphasic motor unit action potentials of a short duration. Genetic analysis revealed a heterozygous mutation in CAPN3, suggestive of autosomal dominant calpainopathy or limb girdle muscular dystrophy. He was administered intravenous potassium and magnesium supplements, followed by oral potassium chloride, magnesium oxide, and potassium-sparing diuretics. The patient had improved muscle strength on follow-up, with resolution of the electrolyte abnormalities. This case report highlights this rare clinical entity, its variable clinical manifestations, and the pathophysiological mechanisms involved in electrolyte abnormalities.

摘要

成人起病的伴有钙蛋白酶病的吉特曼综合征在糖尿病患者中是一种罕见的临床病症。我们报告一例52岁男性糖尿病患者,该患者表现为肌肉无力和疲劳。经评估,他大腿和骨盆带肌肉力量减弱。实验室检查显示低钾血症、低镁血症、代谢性碱中毒、尿钾增多和尿钙减少,这些结果导致诊断为吉特曼综合征。肌电图显示为肌病模式,运动单位动作电位呈短时限多相性。基因分析显示CAPN3存在杂合突变,提示常染色体显性钙蛋白酶病或肢带型肌营养不良。给予他静脉补充钾和镁,随后口服氯化钾、氧化镁和保钾利尿剂。随访时患者肌肉力量有所改善,电解质异常得到纠正。本病例报告突出了这种罕见的临床实体、其多样的临床表现以及电解质异常所涉及的病理生理机制。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f39b/12163201/286fc23a3832/CRIM2025-4210190.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f39b/12163201/286fc23a3832/CRIM2025-4210190.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f39b/12163201/286fc23a3832/CRIM2025-4210190.001.jpg

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本文引用的文献

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Type 2 diabetes mellitus caused by Gitelman syndrome-related hypokalemia: A case report.吉特曼综合征相关低钾血症所致2型糖尿病:一例报告
Medicine (Baltimore). 2020 Jul 17;99(29):e21123. doi: 10.1097/MD.0000000000021123.
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Clinical variability in calpainopathy: what makes the difference?钙蛋白酶病的临床变异性:差异何在?
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