Freeman Alexandra F, Jacobsohn David A, Shulman Stanford T, Bellini William J, Jaggi Preeti, de Leon Guillermo, Keating Gesina F, Kim Francine, Pachman Lauren M, Kletzel Morris, Duerst Reggie E
Division of Infectious Disease, Department of Pediatrics, Northwestern University Feinberg School of Medicine and Children's Memorial Hospital, 2300 Children's Plaza, Chicago, IL 60614, USA.
Pediatrics. 2004 Nov;114(5):e657-60. doi: 10.1542/peds.2004-0949.
Measles inclusion body encephalitis (MIBE) is a disease of the immunocompromised host and typically occurs within 1 year of acute measles infection or vaccination. We report a 13-year-old boy who had chronic granulomatous disease and presented 38 days after stem cell transplantation with afebrile focal seizures that progressed despite multiple anticonvulsants. After an extensive diagnostic evaluation, brain biopsy was performed, revealing numerous intranuclear inclusion bodies consistent with paramyxovirus nucleocapsids. Measles studies including reverse transcriptase-polymerase chain reaction and viral growth confirmed measles virus, genotype D3. Immunohistochemistry was positive for measles nucleoprotein. Despite intravenous ribavirin therapy, the patient died. MIBE has not been described in stem cell recipients but is a disease of immunocompromised hosts and typically occurs within 1 year of measles infection, exposure, or vaccination. Our case is unusual as neither the patient nor the stem cell donor had apparent recent measles exposure or vaccination, and neither had recent travel to measles-endemic regions. The patient had an erythematous rash several weeks before the neurologic symptoms; however, skin biopsy was consistent with graft-versus-host disease, and immunohistochemistry studies for measles nucleoprotein were negative. As measles genotype D3 has not been seen in areas where the child lived since his early childhood, the possibility of an unusually long latency period between initial measles infection and MIBE is raised. In addition, this case demonstrates the utility of brain biopsy in the diagnosis of encephalitis of unknown cause in the immunocompromised host.
麻疹包涵体脑炎(MIBE)是免疫功能低下宿主的一种疾病,通常发生在急性麻疹感染或接种疫苗后1年内。我们报告一名13岁男孩,他患有慢性肉芽肿病,在干细胞移植后38天出现无热局灶性癫痫发作,尽管使用了多种抗惊厥药物,病情仍进展。经过广泛的诊断评估后,进行了脑活检,发现大量与副粘病毒核衣壳一致的核内包涵体。包括逆转录聚合酶链反应和病毒培养在内的麻疹研究证实为麻疹病毒,基因型为D3。免疫组织化学检测麻疹核蛋白呈阳性。尽管给予了静脉注射利巴韦林治疗,患者仍死亡。MIBE在干细胞接受者中尚未见报道,但它是免疫功能低下宿主的一种疾病,通常发生在麻疹感染、接触或接种疫苗后1年内。我们的病例不同寻常,因为患者和干细胞供体近期均无明显的麻疹接触或接种史,且近期均未前往麻疹流行地区。患者在出现神经症状前几周有红斑皮疹;然而,皮肤活检符合移植物抗宿主病,麻疹核蛋白免疫组织化学研究为阴性。由于自幼儿期起该患儿居住地区未见麻疹基因型D3,因此提出了初始麻疹感染与MIBE之间潜伏期异常延长的可能性。此外,该病例证明了脑活检在免疫功能低下宿主不明原因脑炎诊断中的作用。