Boztug Heidrun, Karitnig-Weiß Cäcilia, Ausserer Bernd, Renner Ellen D, Albert Michael H, Sawalle-Belohradsky Julie, Belohradsky Bernd H, Mann Georg, Horcher Ernst, Rümmele-Waibel Alexandra, Geyeregger Rene, Lakatos Karoly, Peters Christina, Lawitschka Anita, Matthes-Martin Susanne
Department of Paediatrics, St Anna Kinderspital and Children's Cancer Research Institute, Medical University of Vienna, Vienna, Austria.
Pediatr Hematol Oncol. 2012 Oct;29(7):585-94. doi: 10.3109/08880018.2012.714844. Epub 2012 Aug 16.
Dedicator of cytokinesis 8 protein (DOCK8) deficiency is a combined immunodeficiency disorder characterized by an expanding clinical picture with typical features of recurrent respiratory or gastrointestinal tract infections, atopic eczema, food allergies, chronic viral infections of the skin, and blood eosinophilia often accompanied by elevated serum IgE levels. The only definitive treatment option is allogeneic hematopoietic stem cell transplantation (HSCT). We report a patient with early severe manifestation of DOCK8 deficiency, who underwent unrelated allogeneic HSCT at the age of 3 years following a reduced toxicity conditioning regimen. The transplant course was complicated by pulmonary aspergilloma pretransplantation, adenovirus (ADV) reactivation, and cytomegalovirus (CMV) pneumonitis 4 weeks after transplantation. With antifungal and antiviral treatment the patient recovered. Seven months after transplantation the patient is in excellent clinical condition. Eczematous rash, chronic viral skin infections, and food allergies have subsided, associated with normalization of IgE levels and absolute numbers of eosinophils. Chimerism analysis shows stable full donor chimerism. DOCK8 deficiency can be successfully cured by allogeneic HSCT. This treatment option should be considered early after diagnosis, as opportunistic infections and malignancies that occur more frequently during the natural course of the disease are associated with higher morbidity and mortality.
细胞分裂素8蛋白(DOCK8)缺乏症是一种联合免疫缺陷疾病,其临床症状不断扩展,具有反复呼吸道或胃肠道感染、特应性湿疹、食物过敏、皮肤慢性病毒感染以及血液嗜酸性粒细胞增多等典型特征,常伴有血清IgE水平升高。唯一的确定性治疗选择是异基因造血干细胞移植(HSCT)。我们报告了一名DOCK8缺乏症早期严重表现的患者,该患者在3岁时接受了毒性降低的预处理方案后进行了无关供体异基因HSCT。移植过程因移植前肺曲菌球、腺病毒(ADV)再激活以及移植后4周的巨细胞病毒(CMV)肺炎而复杂化。经过抗真菌和抗病毒治疗,患者康复。移植后7个月,患者临床状况良好。湿疹性皮疹、慢性病毒皮肤感染和食物过敏已消退,同时IgE水平和嗜酸性粒细胞绝对数量恢复正常。嵌合体分析显示稳定的完全供体嵌合体。异基因HSCT可成功治愈DOCK8缺乏症。诊断后应尽早考虑这种治疗选择,因为在疾病自然病程中更频繁发生的机会性感染和恶性肿瘤与更高的发病率和死亡率相关。