Writzl Karin, Cale Catherine M, Pierce Christine M, Wilson Louise C, Hennekam Raoul C M
Department of Clinical Genetics, Great Ormond Street Hospital for Children, London, UK.
Eur J Med Genet. 2007 Sep-Oct;50(5):338-45. doi: 10.1016/j.ejmg.2007.05.002. Epub 2007 May 27.
Immune deficiency can be part of CHARGE syndrome but often receives only limited attention. We present two patients with CHARGE syndrome confirmed CHD7 mutations who had severe T-cell deficiency, and review 15 CHARGE patients from the literature with immunological problems. Most of them had severe T-cell deficiency, although the spectrum also included mild T-cell deficiency and isolated humoral immune deficiency. We conclude that immunodeficiency can form an important symptom in CHARGE syndrome although the frequency and exact nature are still insufficiently known. We propose to evaluate immune functions in all CHARGE syndrome patients, to estimate the frequency and nature of the accompanying immunodeficiency, and to obtain better data regarding prognosis and management.
免疫缺陷可能是CHARGE综合征的一部分,但往往仅受到有限关注。我们报告了两名经证实存在CHD7突变的CHARGE综合征患者,他们患有严重的T细胞缺陷,并回顾了文献中15例有免疫问题的CHARGE患者。他们大多数有严重的T细胞缺陷,不过其范围也包括轻度T细胞缺陷和孤立性体液免疫缺陷。我们得出结论,免疫缺陷可成为CHARGE综合征的一个重要症状,尽管其发生率和确切性质仍了解不足。我们建议对所有CHARGE综合征患者评估免疫功能,以估计伴随免疫缺陷的发生率和性质,并获取关于预后和管理的更好数据。