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免疫缺陷、着丝粒不稳定和面部异常综合征(ICF)患儿的临床表现和免疫表型存在可变性。

Immunodeficiency, Centromeric Region Instability, and Facial Anomalies Syndrome (ICF) in a Boy with Variable Clinical and Immunological Presentations.

机构信息

Department of Allergy and Clinical Immunology, Iran University of Medical Sciences, Tehran, Iran.

Pediatric Growth and Development Research Center, Iran University of Medical Sciences, Tehran, Iran.

出版信息

Iran J Allergy Asthma Immunol. 2021 Apr 17;20(2):249-254.

PMID:33904683
Abstract

Immunodeficiency, centromeric instability, and facial anomalies (ICF) syndrome is a rare primary immunodeficiency disorder characterized by recurrent infections and low immunoglobulin levels due to variable combined immunodeficiency, and centromeric region instability, and facial dysmorphism. We describe a 12-year-old boy with recurrent respiratory tract infections, facial anomalies, scoliosis, and psychomotor retardation. He had recurrent pneumonia with low serum IgG and IgM levels during infancy and preschool age. Later at the age of 10, he developed recurrent ear infections. An IgA and IgM deficiency was found accompanied by a normal B-cell and T-cell count as well as an impaired candida-induced T-cell proliferation. Further evaluations revealed a missense mutation in the DNMT3B gene on chromosome 20. Chromosomal analysis showed a sunburst multi-radial feature on chromosome 1, which is a hallmark of ICF syndrome. The genetic mutation and chromosomal abnormality along with clinical findings are compatible with the diagnosis of ICF syndrome. To the best of our knowledge, this is the first time that scoliosis is observed in an ICF patient. The additional variable clinical symptoms in the case were the presence of spastic gait as well as hypogammaglobulinemia with immunoglobulin isotype switch at different ages.

摘要

免疫缺陷、着丝粒不稳定和面部异常(ICF)综合征是一种罕见的原发性免疫缺陷病,其特征为由于可变的联合免疫缺陷、着丝粒区不稳定和面部发育不良导致的复发性感染和低免疫球蛋白水平。我们描述了一名 12 岁男孩,他反复出现呼吸道感染、面部异常、脊柱侧凸和精神运动发育迟缓。他在婴儿期和学龄前反复发生肺炎,伴有低血清 IgG 和 IgM 水平。后来,在 10 岁时,他出现了反复耳部感染。发现存在 IgA 和 IgM 缺陷,同时伴有正常的 B 细胞和 T 细胞计数以及受损的念珠菌诱导的 T 细胞增殖。进一步评估发现,20 号染色体上的 DNMT3B 基因存在错义突变。染色体分析显示 1 号染色体上存在四射状多辐射特征,这是 ICF 综合征的标志。基因突变和染色体异常以及临床发现与 ICF 综合征的诊断相符。据我们所知,这是首次在 ICF 患者中观察到脊柱侧凸。该病例的其他可变临床症状为存在痉挛性步态以及不同年龄段的低丙种球蛋白血症和免疫球蛋白同种型转换。

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引用本文的文献

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Immunodeficiency, centromeric instability, and facial anomalies (ICF) syndrome identified by whole-exome sequencing (WES): a case report from a developing country.通过全外显子组测序(WES)鉴定的免疫缺陷、着丝粒不稳定和面部异常(ICF)综合征:来自一个发展中国家的病例报告
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