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血清阴性重症肌无力,一种特纳综合征中的罕见自身免疫性疾病。

Seronegative Myasthenia Gravis, as a Rare Autoimmune Condition in Turner Syndrome.

作者信息

Sharief Rimah, Miodovnik Amir, Motaghedi Roja

机构信息

Infant and Children's Hospital of Brooklyn, Maimonides Medical Center, 977 48th Street, Brooklyn, NY 11219, USA.

出版信息

Case Rep Pediatr. 2017;2017:6863938. doi: 10.1155/2017/6863938. Epub 2017 Nov 14.

Abstract

Girls with Turner syndrome (TS), especially with isochromosome 46,X,i(X)(q10), are prone to develop autoimmunity. Associations of several autoimmune conditions with TS have been frequently described in the past. However, the unique combination of TS and myasthenia gravis (MG) has been reported only once before in a girl with mosaic monosomy 45,X/46,XX. Here, we present the second case of a girl affected with seronegative MG but with mosaic isochromosome TS. This is a child with developmental delay presented with muscle weakness, frequent fall, and bilateral ptosis. Diagnosis of MG was made based on positive Tensilon and electromyography tests and excellent response to intravenous immunoglobulin. At the age of 11 years due to short stature and developmental delay, a karyotype was done and revealed the mosaic isochromosome 45,X/46,X,i(X)(q10). Overall, clinicians should be aware of the vulnerability of girls with TS to autoimmunity, especially if the isochromosome 46,X,i(X)(q10) karyotype is identified. Furthermore, if a child with TS develops muscle weakness, ptosis, or ophthalmoplegia, MG should also be included in the differential diagnosis, particularly if other concurrent autoimmune conditions are present.

摘要

患有特纳综合征(TS)的女孩,尤其是具有46,X,i(X)(q10)等臂染色体的女孩,容易发生自身免疫。过去曾频繁描述过几种自身免疫性疾病与TS的关联。然而,TS与重症肌无力(MG)的独特组合此前仅在一名患有45,X/46,XX嵌合型单体的女孩中被报道过一次。在此,我们报告第二例患有血清阴性MG但伴有嵌合型等臂染色体TS的女孩。这是一名发育迟缓的儿童,表现为肌肉无力、频繁跌倒和双侧上睑下垂。基于腾喜龙试验阳性和肌电图检查以及对静脉注射免疫球蛋白的良好反应,诊断为MG。11岁时,由于身材矮小和发育迟缓,进行了核型分析,结果显示为45,X/46,X,i(X)(q10)嵌合型等臂染色体。总体而言,临床医生应意识到患有TS的女孩易患自身免疫性疾病,尤其是在鉴定出46,X,i(X)(q10)核型时。此外,如果患有TS的儿童出现肌肉无力、上睑下垂或眼肌麻痹,MG也应纳入鉴别诊断,特别是在存在其他并发自身免疫性疾病的情况下。

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