Sato Yutaka, Mishimagi Takashi, Katsuki Yuko, Harada Kiyoshi
Assistant Professor, Division of Maxillofacial Surgery, Department of Maxillofacial and Neck Reconstruction, Faculty of Medical and Dental Sciences, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
Part-Time Instructor, Division of Maxillofacial Surgery, Department of Maxillofacial and Neck Reconstruction, Faculty of Medical and Dental Sciences, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.
J Oral Maxillofac Surg. 2014 Jul;72(7):1396.e1-7. doi: 10.1016/j.joms.2013.12.034. Epub 2014 Jan 15.
X-linked agammaglobulinemia (XLA) is a congenital immune deficiency disorder caused by abnormal antibody production. It is a rare disease with an estimated frequency of 1 in 379,000 that has X-linked recessive heredity and develops only in males. The clinical problems include bacterial infection such as otitis media, sinusitis, and bronchitis. In recent years it has become possible to diagnose XLA in the early stage and intravenous immunoglobulin replacement therapy has permitted survival to adulthood. However, there have been no reports of oral surgery in patients with XLA. Here, we describe a case in which immunoglobulin replacement therapy given pre- and postoperatively was used to control infection in oral surgery and maxillary distraction osteogenesis performed for improving occlusion and appearance of a cleft lip and palate in a patient with XLA.
X连锁无丙种球蛋白血症(XLA)是一种由抗体产生异常引起的先天性免疫缺陷疾病。它是一种罕见疾病,估计发病率为1/379,000,具有X连锁隐性遗传,仅在男性中发病。临床问题包括中耳炎、鼻窦炎和支气管炎等细菌感染。近年来,早期诊断XLA已成为可能,静脉注射免疫球蛋白替代疗法使患者能够存活至成年。然而,尚无关于XLA患者口腔手术的报道。在此,我们描述了一例XLA患者,术前和术后给予免疫球蛋白替代疗法以控制口腔手术中的感染,并进行上颌骨牵张成骨术以改善唇腭裂患者的咬合和外观。