Zheng Xiao-Yu, Xu Jia, Li Wei, Li Si-Si, Shi Cai-Ping, Zhao Zheng-Yan, Mao Jian-Hua, Chen Xi
From the Department of Ophthalmology (X-YZ, C-PS); Department of Hematology-Oncology (S-SL); Department of Child Health Care (Z-YZ); Department of Nephrology (J-HM); the Central Lab (WL, XC), The Children's Hospital of Zhejiang University School of Medicine, Hangzhou; and Department of Ophthalmology (JX), the Second Affiliated Hospital of Zhejiang University School of Medicine, Hangzhou, Zhejiang, China.
Medicine (Baltimore). 2016 Mar;95(12):e3106. doi: 10.1097/MD.0000000000003106.
Dyskeratosis congenita (DC) is an inherited bone marrow failure syndrome, usually presented with abnormal skin pigmentation, nail dystrophy, and oral leukoplakia. The main cause of mortality in DC is immunodeficiency and vital infection. DC involves multisystem, but retinal involvements are rare.Herein, we report an unusual case of pediatric DC suffering from frosted branch angiitis (FBA) after recovery of mycoplasma pneumonia. Cytomegalovirus infection and cytokine changes were found relevant to the onset of FBA. Despite corticosteroids, antiviral medication, and hematopoietic stem cell transplantation, the patient ended in poor vision with optic atrophy.This case implies that pediatricians should be aware of FBA as a rare retinal manifestation in children with DC and bone marrow failure. Cytomegalovirus may be one of the common causes and cytokines could be triggering factors.
先天性角化不良(DC)是一种遗传性骨髓衰竭综合征,通常表现为皮肤色素沉着异常、指甲营养不良和口腔白斑。DC患者的主要死亡原因是免疫缺陷和严重感染。DC累及多系统,但视网膜受累罕见。在此,我们报告一例特殊的儿童DC病例,该患儿在支原体肺炎康复后患上了霜枝样视网膜血管炎(FBA)。发现巨细胞病毒感染和细胞因子变化与FBA的发病有关。尽管使用了皮质类固醇、抗病毒药物和造血干细胞移植,该患者最终视力不佳并出现视神经萎缩。该病例提示儿科医生应注意FBA是DC和骨髓衰竭患儿罕见的视网膜表现。巨细胞病毒可能是常见病因之一,细胞因子可能是触发因素。