当前位置: X-MOL 学术Horm. Res. Paediatr. › 论文详情
Our official English website, www.x-mol.net, welcomes your feedback! (Note: you will need to create a separate account there.)
New onset Primary Adrenal Insufficiency and Autoimmune Hypothyroidism in a Pediatric Patient presenting with MIS-C
Hormone Research in Paediatrics ( IF 2.6 ) Pub Date : 2022-05-24 , DOI: 10.1159/000525227
Myrto Eleni Flokas 1 , Victoria H Bustamante 1 , Roopa Kanakatti Shankar 1, 2
Affiliation  

Introduction: There is emerging speculation that the inflammatory state associated with SARS-CoV-2 infection may trigger autoimmune conditions, but no causal link is established. There are reports of autoimmune thyroiditis, and adrenal insufficiency in adults post-COVID-19. We describe the first pediatric report of autoimmune adrenal insufficiency and hypothyroidism after COVID-19. Case Presentation: A 14-year-old previously healthy girl, with vitiligo, presented in shock following one-week of fever, lethargy, diarrhea, and vomiting. Three weeks prior, she had congestion and fatigue, and known familial exposure for COVID-19. Labs were remarkable for sodium 129mmol/L, K 4.3mmol/L, creatinine 2.9mg/dL, hemoglobin 8.3 g/dL and positive COVID-19 PCR and SARS CoV-2 IgG. She was resuscitated with normal saline and required pressor support. EKG showed abnormal repolarization presumed secondary to myocarditis. She met criteria for Multisystem Inflammatory Syndrome in Children (MIS-C), received intravenous immune globulin and IL-1R antagonist, and was admitted for intensive care. Persistent hypotension despite improved inflammatory markers, and an undetectable cortisol led to initiation of hydrocortisone. She was then able to rapidly wean off vasopressors and hydrocortisone within 48 hours. Thereafter, tests undertaken for persistent bradycardia confirmed autoimmune hypothyroidism with TSH 131mcunit/ml, freeT4 0.85ng/dL, and positive thyroid autoantibodies. Basal and stimulated cortisol were <1 mcg/dL on a standard 250mcg cosyntropin stimulation test, with baseline ACTH >1250pg/mL confirming primary adrenal insufficiency. Treatment was initiated with hydrocortisone, levothyroxine, and fludrocortisone. Adrenal sonogram did not reveal any hemorrhage and anti-adrenal antibody titers were positive. The family retrospectively reported oligomenorrhea, increased salt craving in the months prior, and family history of autoimmune thyroiditis. The cytokine panel was notably different from other cases of MIS-C. Conclusion: This is the first pediatric report, to our knowledge, of primary adrenal insufficiency and hypothyroidism following COVID-19, leading to a unique presentation of autoimmune polyglandular syndrome type 2. The initial presentation was attributed to MIS-C, but the subsequent clinical course suggests the possibility of adrenal crisis. It remains unknown if COVID-19 had a causal relationship in triggering the autoimmune adrenal insufficiency and hypothyroidism.


中文翻译:

患有 MIS-C 的儿科患者的新发原发性肾上腺功能不全和自身免疫性甲状腺功能减退症

简介:有人猜测,与 SARS-CoV-2 感染相关的炎症状态可能会引发自身免疫性疾病,但尚未确定因果关系。有报告称 COVID-19 后成人会出现自身免疫性甲状腺炎和肾上腺功能不全。我们描述了 COVID-19 后自身免疫性肾上腺功能不全和甲状腺功能减退症的第一份儿科报告。病例介绍:一名患有白斑病的 14 岁女孩,在经历一周的发烧、嗜睡、腹泻和呕吐后休克就诊。三周前,她出现充血和疲劳,并且已知有 COVID-19 家族史。实验室对钠 129 毫摩尔/升、钾 4.3 毫摩尔/升、肌酐 2.9 毫克/分升、血红蛋白 8.3 克/分升以及阳性 COVID-19 PCR 和 SARS CoV-2 IgG 非常显着。她用生理盐水复苏并需要升压支持。心电图显示复极化异常,推测继发于心肌炎。她符合儿童多系统炎症综合征 (MIS-C) 的诊断标准,接受了静脉注射免疫球蛋白和 IL-1R 拮抗剂治疗,并入院接受重症监护。尽管炎症标志物有所改善,但仍持续低血压,皮质醇检测不到导致开始使用氢化可的松。然后,她能够在 48 小时内迅速停用血管加压药和氢化可的松。此后,针对持续性心动过缓进行的测试证实自身免疫性甲状腺功能减退症,TSH 131mcunit/ml,freeT4 0.85ng/dL,甲状腺自身抗体阳性。在标准的 250mcg 促肾上腺皮质激素刺激试验中,基础和刺激皮质醇 <1 mcg/dL,基线 ACTH >1250pg/mL 证实原发性肾上腺功能不全。治疗开始于氢化可的松,左甲状腺素和氟氢可的松。肾上腺超声检查未发现任何出血,抗肾上腺抗体滴度呈阳性。该家族回顾性地报告了月经稀发、前几个月对盐的渴望增加以及自身免疫性甲状腺炎的家族史。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。肾上腺超声检查未发现任何出血,抗肾上腺抗体滴度呈阳性。该家族回顾性地报告了月经稀发、前几个月对盐的渴望增加以及自身免疫性甲状腺炎的家族史。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。肾上腺超声检查未发现任何出血,抗肾上腺抗体滴度呈阳性。该家族回顾性地报告了月经稀发、前几个月对盐的渴望增加以及自身免疫性甲状腺炎的家族史。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。该家族回顾性地报告了月经稀发、前几个月对盐的渴望增加以及自身免疫性甲状腺炎的家族史。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。该家族回顾性地报告了月经稀发、前几个月对盐的渴望增加以及自身免疫性甲状腺炎的家族史。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。细胞因子组与其他 MIS-C 病例明显不同。结论:据我们所知,这是第一份关于 COVID-19 后原发性肾上腺功能不全和甲状腺功能减退的儿科报告,导致 2 型自身免疫性多腺体综合征的独特表现。最初的表现归因于 MIS-C,但随后的临床表现当然提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。但随后的临床过程提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。但随后的临床过程提示肾上腺危象的可能性。目前尚不清楚 COVID-19 是否与引发自身免疫性肾上腺功能不全和甲状腺功能减退症有因果关系。
更新日期:2022-05-25
down
wechat
bug