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15 岁男孩中同时出现 Addison 病和 Graves 病的 II 型自身免疫性多腺体综合征:病例报告及文献复习。

Autoimmune polyglandular syndrome type II with co-manifestation of Addison's and Graves' disease in a 15-year-old boy: case report and literature review.

机构信息

Catholic Childrens Hospital Wilhelmstift, Department of Paediatrics, Hamburg, Germany.

出版信息

J Pediatr Endocrinol Metab. 2020 Apr 28;33(4):575-578. doi: 10.1515/jpem-2019-0506.

Abstract

Background Autoimmune polyglandular syndrome type II (APS II) is defined as the combination of autoimmune adrenal insufficiency and autoimmune thyroid disease (AITD) and/or type I diabetes mellitus (T1DM) in the same patient. Case presentation A 15-year-old boy had a history of weight loss, nausea and vomiting, headache, restlessness, and tanned skin. He was diagnosed with Graves' disease. Two weeks after carbimazol therapy was commenced, he presented with adrenal crises (fever, arterial hypotension, hyponatremia, adrenocorticotropic hormone [ACTH] 1119.6 ng/mL [normal range <60] and low cortisol). He received hydrocortisone and fludrocortisone, and improved quickly. Thyroid-stimulating hormone (TSH) receptor autoantibodies as well as 21-hydroxylase antibodies were elevated. Conclusions Although the combination of Graves' and Addison's disease is extremely rare, especially in children, it is critical to make the diagnosis. Accelerated metabolic rate increased the risk for adrenal crises in our patient. This case contributes to the spectrum of APS II and its manifestation.

摘要

背景 自身免疫性多腺体综合征 II 型(APS II)定义为同一患者同时存在自身免疫性肾上腺功能不全、自身免疫性甲状腺疾病(AITD)和/或 1 型糖尿病(T1DM)。

病例介绍 一名 15 岁男孩有体重减轻、恶心呕吐、头痛、烦躁和皮肤黝黑的病史。他被诊断为格雷夫斯病。在开始使用甲巯咪唑治疗两周后,他出现了肾上腺危象(发热、动脉低血压、低钠血症、促肾上腺皮质激素 [ACTH] 1119.6ng/mL[正常范围<60]和低皮质醇)。他接受了氢化可的松和氟氢可的松治疗,很快得到改善。促甲状腺激素(TSH)受体自身抗体和 21-羟化酶抗体升高。

结论 尽管格雷夫斯病和艾迪生病同时发生的情况极其罕见,尤其是在儿童中,但明确诊断至关重要。快速的代谢率增加了我们患者发生肾上腺危象的风险。该病例丰富了 APS II 及其表现谱。

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