Aricò M, Bettinelli A, Maccario R, Clementi R, Bossi G, Danesino C
Clinica Pediatrica, IRCCS Policlinico S. Matteo, Pavia, Italy.
Am J Med Genet. 1999 Dec 3;87(4):329-30.
We report on a new patient with deletion of 22q11 associated with hemophagocytic lymphohistiocytosis and a fatal outcome. She had minor facial anomalies and cardiac malformation corresponding to those described in del (22q11) syndrome, normal T and B cell function and NK activity; bone marrow aspiration showed active erythrophagocytosis. Our case in addition to two other children reported previously suggest that such a rare association between lymphocyte-macrophage activation and deletion of 22q11 may be more frequent than previously recognized.
我们报告了一名患有22q11缺失且伴有噬血细胞性淋巴组织细胞增生症及致命结局的新患者。她有轻微面部异常和与del(22q11)综合征中所描述相符的心脏畸形,T细胞和B细胞功能及NK活性正常;骨髓穿刺显示有活跃的红细胞吞噬现象。我们的病例以及之前报道的另外两名儿童提示,淋巴细胞-巨噬细胞激活与22q11缺失之间这种罕见的关联可能比之前认为的更为常见。