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1型自身免疫性多腺体综合征在五十多岁时的非典型表现。

Atypical presentation of autoimmune polyglandular syndrome type 1 in the fifth decade.

作者信息

Sanjeevi Aditya, Asirvatham Adlyne Reena, Balachandran Karthik, Mahadevan Shriraam

机构信息

Internal Medicine, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India

Endocrinology, Sri Ramachandra University Medical College, Chennai, Tamil Nadu, India.

出版信息

BMJ Case Rep. 2021 Apr 26;14(4):e241680. doi: 10.1136/bcr-2021-241680.

Abstract

A 45-year-old woman presented to us with a short-term history of nausea, vomiting and giddiness. On arrival at our hospital, examination revealed postural hypotension. Fluid resuscitation with intravenous normal saline was commenced. She also had chronic mucocutaneous candidiasis and nail changes suggestive of ectodermal dystrophy. Detailed history taking revealed that she had never attained menarche. Serum biochemistries showed hyponatraemia, hyperkalaemia, and hypocalcaemia (sodium, 127 mEq/L; potassium, 6 mEq/L; and albumin-corrected calcium, 6 mg/dL). Adrenocorticotropic hormone-stimulated cortisol (16.7 mcg/dL) was suboptimal favouring adrenal insufficiency. She was started on hydrocortisone and fludrocortisone supplementation. Additionally, the parathyroid hormone was inappropriately low (3.8 pg/mL) confirming hypoparathyroidism. Oral calcium and active vitamin D supplementation were added. With the above clinical and biochemical picture, namely, clustering of primary amenorrhoea, adrenal insufficiency and hypoparathyroidism, the diagnosis pointed towards autoimmune polyglandular syndrome. Genetic workup revealed a deletion in exon 8 of the autoimmune regulator gene confirming the diagnosis of autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy/autoimmune polyglandular syndrome type 1 .

摘要

一名45岁女性因短期出现恶心、呕吐和头晕症状前来就诊。入院时检查发现有体位性低血压。开始静脉输注生理盐水进行液体复苏。她还患有慢性黏膜皮肤念珠菌病以及提示外胚层发育不良的指甲改变。详细询问病史发现她从未月经初潮。血清生化检查显示低钠血症、高钾血症和低钙血症(钠,127 mEq/L;钾,6 mEq/L;白蛋白校正钙,6 mg/dL)。促肾上腺皮质激素刺激后的皮质醇水平(16.7 mcg/dL)欠佳,提示肾上腺功能不全。开始给予氢化可的松和氟氢可的松补充治疗。此外,甲状旁腺激素水平异常低(3.8 pg/mL),确诊为甲状旁腺功能减退。加用口服钙剂和活性维生素D补充治疗。结合上述临床和生化表现,即原发性闭经、肾上腺功能不全和甲状旁腺功能减退聚集出现,诊断指向自身免疫性多腺体综合征。基因检测发现自身免疫调节基因第8外显子缺失,确诊为自身免疫性多内分泌腺病-念珠菌病-外胚层发育不良/1型自身免疫性多腺体综合征。

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本文引用的文献

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Varying presentation of type 1 polyglandular failure in India.印度1型多腺体功能减退的不同表现
J Pediatr Endocrinol Metab. 2010 Mar;23(3):271-6. doi: 10.1515/jpem.2010.23.3.271.

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