Amir N, Chemke J, Shapira Y
Pediatric Department, Barzilai Medical Center, Ashkelon, Israel.
Pediatr Neurol. 1985 Jul-Aug;1(4):249-51. doi: 10.1016/s0887-8994(85)80011-4.
An infant presented with congenital hypotonia, poor sucking, apathy, and areflexia. Muscle biopsy at two months of age revealed numerous nemaline rods, suggesting congenital nemaline myopathy. During the ensuing months, familial dysautonomia was suggested by recurrent pulmonary infections, dysphagia, alacrima, hyperhydrosis, emotional lability, and unexplained episodes of hyperthermia and breath-holding spells. The diagnosis was confirmed by positive intradermal histamine and ocular mecholyl tests. The finding of nemaline rods adds a new facet to the recognized polymorphic presentation of familial dysautonomia.
一名婴儿出现先天性肌张力减退、吸吮无力、淡漠和反射消失。两个月大时的肌肉活检显示有大量杆状体,提示先天性杆状体肌病。在随后的几个月里,反复出现的肺部感染、吞咽困难、无泪、多汗、情绪不稳定以及不明原因的高热和屏气发作提示为家族性自主神经功能障碍。皮内组胺试验和眼用乙酰甲胆碱试验阳性证实了诊断。杆状体的发现为家族性自主神经功能障碍公认的多形性表现增添了新的方面。