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法布里病患者的左心室心肌致密化不全:过度诊断、法布里病的形态学表现还是同一患者中两种不相关的罕见病症?

Left ventricular noncompaction in a patient with fabry disease: overdiagnosis, morphological manifestation of fabry disease or two unrelated rare conditions in the same patient?

作者信息

Azevedo Olga, Gaspar Paulo, Sá Miranda Clara, Cunha Damião, Medeiros Rosa, Lourenço António

机构信息

Cardiology Department, Centro Hospitalar do Alto Ave, Guimarães, Portugal. olgazevedo @ yahoo.com.br

出版信息

Cardiology. 2011;119(3):155-9. doi: 10.1159/000330924. Epub 2011 Sep 23.

DOI:10.1159/000330924
PMID:21952291
Abstract

We report a clinical case of a young female with Fabry disease but without left ventricular hypertrophy, which fulfills the diagnostic criteria of left ventricular noncompaction (LVNC). To our knowledge, this is the first report of LVNC in a patient with Fabry disease. The possibility of an overdiagnosis of LVNC is discussed based on the limitations of the current diagnostic criteria. This case was further investigated by genetic analysis, which came to demonstrate the limited usefulness of genetic testing in the diagnosis of LVNC. Assuming a true trabecular pattern of LVNC, the hypothesis that the same patient has two unrelated and rare conditions, although possible, is unlikely. The genetic and clinical heterogeneity of LVNC is discussed and supports, along with this clinical case, the hypothesis that LVNC is a morphological expression of different diseases rather than a distinct cardiomyopathy. Accordingly, LVNC could be a rare cardiac manifestation of Fabry disease.

摘要

我们报告了一例年轻女性法布里病患者的临床病例,该患者无左心室肥厚,但符合左心室心肌致密化不全(LVNC)的诊断标准。据我们所知,这是法布里病患者中LVNC的首例报告。基于当前诊断标准的局限性,讨论了LVNC过度诊断的可能性。通过基因分析对该病例进行了进一步研究,结果表明基因检测在LVNC诊断中的作用有限。假设LVNC的小梁模式为真性,同一患者患有两种不相关的罕见疾病这一假设虽然有可能,但可能性不大。讨论了LVNC的基因和临床异质性,连同该临床病例一起支持了LVNC是不同疾病的形态学表现而非一种独特心肌病的假设。因此,LVNC可能是法布里病罕见的心脏表现。

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