Buescher E S, Gaither T, Nath J, Gallin J I
Blood. 1985 Jun;65(6):1382-90.
We evaluated a 3-year-old female patient with leukocytosis, recurrent infections, severe periodontal disease, and a history of delayed separation of the umbilical stump. This patient's polymorphonuclear leukocytes (PMNs) had normal membrane depolarization responses, normal oxygen metabolism, normal granule secretion responses, normal bactericidal activity, and normal C3b rosetting. However, by fluorescent cell analysis and C3bi rosetting, it was determined that her cells lacked the C3bi receptor. In addition, the patient's PMNs showed markedly abnormal chemotaxis, adherence, and aggregation responses, and partial abnormalities were detected in PMN spreading and phagocytosis. Sodium dodecyl sulfate-polyacrylamide gel electrophoresis revealed that the subject's neutrophil cytoplasts were missing a 180,000-dalton moiety. Her monocytes also had defective chemotaxis and failed to adhere and grow normally in culture. Epstein-Barr virus-transformed B cells from the patient lacked an aggregation response to phorbol myristate acetate. Laboratory and clinical evaluations of this patient's mother showed no abnormalities. These studies demonstrate that C3bi receptor deficiency can be associated with functional abnormalities in multiple myeloid cells and that the absence of C3bi receptor is associated with abnormal adherence-related functions of these cells.
我们评估了一名3岁女性患者,她患有白细胞增多症、反复感染、严重的牙周疾病,并有脐带残端延迟脱落史。该患者的多形核白细胞(PMN)具有正常的膜去极化反应、正常的氧代谢、正常的颗粒分泌反应、正常的杀菌活性和正常的C3b玫瑰花结形成。然而,通过荧光细胞分析和C3bi玫瑰花结形成检测发现,她的细胞缺乏C3bi受体。此外,患者的PMN表现出明显异常的趋化性、黏附性和聚集反应,并且在PMN铺展和吞噬作用中检测到部分异常。十二烷基硫酸钠 - 聚丙烯酰胺凝胶电泳显示,该患者的中性粒细胞胞质体缺少一个180,000道尔顿的部分。她的单核细胞也具有趋化性缺陷,并且在培养中不能正常黏附并生长。该患者母亲的实验室和临床评估未显示异常。这些研究表明,C3bi受体缺陷可能与多种髓样细胞的功能异常有关,并且C3bi受体的缺失与这些细胞的异常黏附相关功能有关。