Kearney D L, Titus J L, Hawkins E P, Ott D A, Garson A
Circulation. 1987 Apr;75(4):705-10. doi: 10.1161/01.cir.75.4.705.
We have observed in 11 infants, aged 2 years or less, a distinct clinicopathologic lesion responsible for tachyarrhythmias that were fatal in 96% (25/26) of previously reported cases. Nine of the 11 patients, who underwent electrophysiologic mapping and surgical excision of the lesion, have survived, with follow-up periods ranging from 1 month to 6 years. The morphologic findings in these 11 patients and in the 26 cases cited in the literature are reviewed. Pathogenic considerations have included viral-induced lesion, cardiomyopathy, neoplasm, and developmental disorder of Purkinje cells. We believe this lesion to be a myocardial hamartoma. Supportive evidence includes prevalence in infants, tumorlike growth pattern without mitotic figures, and association of other developmental abnormalities. Through electrophysiologic mapping, this myocardial hamartoma is potentially accessible to surgical excision and long-term cure.
我们在11名2岁及以下的婴儿中观察到一种独特的临床病理病变,该病变可导致快速性心律失常,在先前报道的病例中,96%(25/26)的此类病例是致命的。11名患者中有9名接受了病变的电生理标测和手术切除,目前均存活,随访时间为1个月至6年。本文回顾了这11名患者以及文献中引用的26例病例的形态学发现。病因学考虑因素包括病毒感染所致病变、心肌病、肿瘤和浦肯野细胞发育障碍。我们认为这种病变是心肌错构瘤。支持这一观点的证据包括其在婴儿中的发病率、无核分裂象的肿瘤样生长模式以及与其他发育异常的关联。通过电生理标测,这种心肌错构瘤有可能通过手术切除并实现长期治愈。