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多发性雀斑样痣患者心血管检查的重要性:两例伴有肥厚型心肌病的豹斑综合征病例

Importance of cardiovascular examination in patients with multiple lentigines: two cases of LEOPARD syndrome with hypertrophic cardiomyopathy.

作者信息

Jurko Tomas, Jurko Alexander, Krsiakova Jana, Jurko Alexander, Minarik Milan, Mestanik Michal

机构信息

a Department of Neonatology, Jessenius Faculty of Medicine in Martin , Comenius University in Bratislava, University Hospital Martin , Martin , Slovakia.

b Pediatric Cardiology Clinic , Martin , Slovakia.

出版信息

Acta Clin Belg. 2019 Apr;74(2):82-85. doi: 10.1080/17843286.2018.1467531. Epub 2018 May 2.

Abstract

Introduction LEOPARD syndrome is a rare genetic disorder characterised by lentigines, electrocardiographic conduction abnormalities, ocular hypertelorism, pulmonary stenosis, abnormal genitalia, retardation of growth and sensorineural deafness. Clinical manifestations are often mild, which may result in difficult and late diagnosis. Cardiac involvement may have a significant impact on the prognosis, however, appearance of severe abnormalities such as hypertrophic cardiomyopathy usually precedes the occurrence of multiple lentigines and may be asymptomatic. Case presentation We report two cases of LEOPARD syndrome with hypertrophic cardiomyopathy in a 10-year-old girl and an 18-year-old boy. In both cases, multiple lentigines, ocular hypertelorism and growth retardation were present. The first patient was followed up at the paediatric cardiology clinic due to the risk of progression of septal hypertrophy and pressure gradient across the left ventricular outflow tract, the second patient underwent surgery for a moderate obstruction of the left ventricular outflow tract with uncomplicated post-operative follow-up. Conclusion In both presented patients, hypertrophic cardiomyopathy was clinically silent and the murmur over the precordium was the sole cardiac abnormality revealed during routine visit. A detailed cardiologic examination should be considered in the patients with suspicion of LEOPARD syndrome since the ventricular hypertrophy is thought to precede the occurrence of lentigines and progress over time.

摘要

引言

豹斑综合征是一种罕见的遗传性疾病,其特征为雀斑、心电图传导异常、眼距增宽、肺动脉狭窄、生殖器异常、生长发育迟缓以及感音神经性耳聋。临床表现通常较为轻微,这可能导致诊断困难且延迟。心脏受累可能对预后产生重大影响,然而,诸如肥厚型心肌病等严重异常通常在多发性雀斑出现之前就已存在,且可能无症状。

病例报告

我们报告了两例豹斑综合征合并肥厚型心肌病的病例,一例为10岁女孩,另一例为18岁男孩。两例均有多发性雀斑、眼距增宽和生长发育迟缓。首例患者因室间隔肥厚进展及左心室流出道压力阶差风险在儿科心脏病诊所接受随访,第二例患者因左心室流出道中度梗阻接受手术,术后随访无并发症。

结论

在这两位患者中,肥厚型心肌病在临床上并无症状,心前区杂音是常规就诊时发现的唯一心脏异常。对于疑似豹斑综合征的患者,应考虑进行详细的心脏检查,因为心室肥厚被认为在雀斑出现之前就已存在且会随时间进展。

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