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一名患有儿童多系统炎症综合征(MIS-C)的儿科患者出现的自身免疫性多内分泌腺病

Autoimmune Polyendocrinopathy in a Pediatric Patient Presenting With Multisystem Inflammatory Syndrome in Children (MIS-C).

作者信息

Sambangi Chaitanya, Collins Patrice, Patel Julisa, Chan Jacqueline

机构信息

Pediatrics, Medical College of Georgia at Augusta University, Augusta, USA.

Pediatric Rheumatology, Children's Hospital of Georgia at Augusta University, Augusta, USA.

出版信息

Cureus. 2023 May 1;15(5):e38407. doi: 10.7759/cureus.38407. eCollection 2023 May.

Abstract

Multisystem inflammatory syndrome (MIS) is a well-known potential sequela of COVID-19 infection. Though prevalence is higher in certain populations, this syndrome is a rare occurrence in children. Beyond MIS, there has been increasing research into COVID infection and the subsequent onset of autoimmune conditions, such as diabetes. However, evidence of a poly-endocrinopathy developing after COVID infection is lacking, and evidence within the pediatric population is virtually nonexistent. In this case, we present the evolution of an autoimmune polyglandular syndrome (APS) type 2 phenotype, consisting of type 1 diabetes, Graves' disease, and adrenal insufficiency, after diagnosis of multisystem inflammatory syndrome of children (MIS-C) in a pediatric patient.  A 15-year-old biracial female without significant past medical history tested positive for COVID-19 and two weeks later presented with respiratory symptoms and other systemic signs. She was admitted for further evaluation and was found to have elevated inflammatory markers, EKG (electrocardiogram) abnormalities, and lab evidence of organ damage. The patient was diagnosed with MIS-C, and treatment was initiated with eventual discharge. One year after this initial visit, the patient returned to the hospital due to weight loss, difficulty breathing, polyuria, polydipsia, nausea, vomiting, and fatigue. A steroid course for MIS-C treatment had been completed three months prior. Exam and lab results confirmed diabetic ketoacidosis (DKA), and the patient was diagnosed with new-onset type 1 diabetes. Further testing determined that she was glutamic acid decarboxylase 65 (GAD-65) positive. DKA was managed in the hospital, and the patient was subsequently discharged with an insulin regimen and endocrine follow-up. A couple of months later, the patient returned to the emergency department (ED) due to two weeks of dyspnea on exertion and dizziness. Since her previous admission for DKA, the patient had contracted COVID-19 again and recovered from her respiratory symptoms. Physical exam and labs were grossly unremarkable; however, the patient had EKG abnormalities and an episode of severe bradycardia, prompting hospitalization. Thyroid workup revealed thyrotoxicosis due to Graves' disease. Due to intermittent hypotension, adrenal labs were obtained. She was found to have adrenal insufficiency as well, with a positive 21-hydroxylase antibody. Throughout these hospitalizations, the patient suffered from skin and hair changes as well, ultimately requiring dermatological intervention.

摘要

多系统炎症综合征(MIS)是新冠病毒感染后一种众所周知的潜在后遗症。尽管在某些人群中的患病率较高,但这种综合征在儿童中较为罕见。除了MIS,关于新冠病毒感染及其后自身免疫性疾病(如糖尿病)发病的研究越来越多。然而,缺乏新冠病毒感染后发生多内分泌腺病的证据,在儿科人群中几乎不存在相关证据。在此病例中,我们报告了一名儿科患者在被诊断为儿童多系统炎症综合征(MIS-C)后,出现的2型自身免疫性多腺体综合征(APS)表型的演变过程,该表型包括1型糖尿病、格雷夫斯病和肾上腺功能不全。一名15岁的混血女性,既往无重大病史,新冠病毒检测呈阳性,两周后出现呼吸道症状和其他全身症状。她因进一步评估入院,发现炎症标志物升高、心电图(EKG)异常以及器官损伤的实验室证据。该患者被诊断为MIS-C,并开始接受治疗,最终出院。首次就诊一年后,患者因体重减轻、呼吸困难、多尿、多饮、恶心、呕吐和疲劳再次入院。三个月前已完成一次针对MIS-C治疗的类固醇疗程。检查和实验室结果证实为糖尿病酮症酸中毒(DKA),患者被诊断为新发1型糖尿病。进一步检测确定她谷氨酸脱羧酶65(GAD-65)呈阳性。DKA在医院得到治疗,患者随后出院并接受胰岛素治疗方案及内分泌随访。几个月后,患者因运动性呼吸困难和头晕两周再次返回急诊科(ED)。自上次因DKA入院以来,该患者再次感染新冠病毒并已从呼吸道症状中康复。体格检查和实验室检查大致正常;然而,患者有心电图异常和一次严重心动过缓发作,促使其住院治疗。甲状腺检查显示因格雷夫斯病导致甲状腺毒症。由于间歇性低血压,进行了肾上腺相关检查。发现她也患有肾上腺功能不全,21-羟化酶抗体呈阳性。在这些住院期间,患者还出现了皮肤和毛发变化,最终需要皮肤科干预。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d77f/10232015/c9d1368fa1e5/cureus-0015-00000038407-i01.jpg

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