Yoshiyama Megumi, Kounami Shinji, Nakayama Keiko, Aoyagi Noriyuki, Yoshikawa Norishige
Department of Pediatrics, Wakayama Medical University, School of Medicine, Wakayama City, Japan.
Pediatr Int. 2008 Aug;50(4):432-5. doi: 10.1111/j.1442-200X.2008.02701.x.
Mycoplasma pneumoniae has been reported to be an etiologic pathogen of infection-associated hemophagocytic lymphohistiocytosis (HLH), but few case reports have been available to date.
The clinical features of four childhood cases of M. pneumoniae-associated hemophagocytic lymphohistiocytosis (Mp-HLH) were retrospectively assessed to obtain data that might be useful for early diagnosis and effective management. The previous English-language literature pertaining to Mp-HLH was also reviewed.
The patients were two boys and two girls, aged between 1 and 11 years of age. One patient was demonstrated to have concurrent infection with rubella. All the patients had typical radiographic features of M. pneumoniae pneumonia, and one patient also had encephalopathy as a complication. All the children underwent bone marrow examination because of antibiotic-refractory fever, mild hepatosplenomegaly, cytopenia, hyperferritinemia and elevated levels of urine beta2-microglobulin. Cytopenia and hepatosplenomegaly in the present patients were relatively mild as compared to those in cases of other infection-associated HLH such as Epstein-Barr virus infection-associated HLH. Treatment with corticosteroids resulted in prompt and complete resolution in two cases, and i.v. high-dose gammaglobulin therapy achieved a complete response in another child. Spontaneous resolution under treatment with antibiotics alone was observed in one case.
Although Mp-HLH is a rare complication of M. pneumoniae infection, it should always be considered in patients with antibiotic-refractory M. pneumoniae infections with cytopenia. Mp-HLH might be effectively treated by corticosteroids or high-dose gammaglobulin. To clarify the diverse clinical manifestations of M. pneumoniae infections, immunological interactions between M. pneumoniae and the host immune system should be further investigated.
据报道,肺炎支原体是感染相关性噬血细胞性淋巴组织细胞增生症(HLH)的病原体,但迄今为止病例报告较少。
回顾性评估4例儿童肺炎支原体相关性噬血细胞性淋巴组织细胞增生症(Mp-HLH)的临床特征,以获取可能有助于早期诊断和有效管理的数据。还查阅了之前关于Mp-HLH的英文文献。
患者为2名男孩和2名女孩,年龄在1至11岁之间。1例患者被证实同时感染风疹。所有患者均有典型的肺炎支原体肺炎影像学特征,1例患者还并发脑病。所有儿童因抗生素治疗无效的发热、轻度肝脾肿大、血细胞减少、高铁蛋白血症和尿β2-微球蛋白水平升高而接受骨髓检查。与其他感染相关性HLH(如爱泼斯坦-巴尔病毒感染相关性HLH)的病例相比,本研究患者的血细胞减少和肝脾肿大相对较轻。2例患者使用皮质类固醇治疗后迅速完全缓解,另1例儿童静脉注射大剂量丙种球蛋白治疗获得完全缓解。1例患者仅用抗生素治疗后自发缓解。
尽管Mp-HLH是肺炎支原体感染的罕见并发症,但对于抗生素治疗无效且伴有血细胞减少的肺炎支原体感染患者,应始终考虑到该病。Mp-HLH可能通过皮质类固醇或大剂量丙种球蛋白有效治疗。为了阐明肺炎支原体感染的各种临床表现,应进一步研究肺炎支原体与宿主免疫系统之间的免疫相互作用。