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四个患有安德森-法布里病的家族中的家族内表型变异性。

Intrafamilial phenotypic variability in four families with Anderson-Fabry disease.

作者信息

Rigoldi M, Concolino D, Morrone A, Pieruzzi F, Ravaglia R, Furlan F, Santus F, Strisciuglio P, Torti G, Parini R

机构信息

Rare Metabolic Diseases Unit, San Gerardo Hospital, Monza, Italy.

出版信息

Clin Genet. 2014 Sep;86(3):258-63. doi: 10.1111/cge.12261. Epub 2013 Sep 20.

Abstract

We analysed the clinical history of 16 hemizygous males affected by Anderson-Fabry Disease, from four families, to verify their intrafamilial phenotypic variability. Seven male patients, ranging from 26 to 61 years of age, died, whereas nine (age range 23-55) are alive. Eleven patients have undergone enzyme replacement therapy (ERT) for a period of 5-10 years. We have found a wide range of intrafamilial phenotypic variability in these families, both in terms of target-organs and severity of the disease. Overall, our findings confirm previous data from the literature showing a high degree of intrafamilial phenotypic variability in patients carrying the same mutation. Furthermore, our results underscore the difficulty in giving accurate prognostic information to patients during genetic counselling, both in terms of rate of disease progression and involvement of different organs, when such prognosis is solely based on the patient's family history.

摘要

我们分析了来自四个家族的16名患有安德森-法布里病的半合子男性的临床病史,以验证其家族内表型变异性。7名年龄在26至61岁之间的男性患者死亡,而9名(年龄范围23 - 55岁)存活。11名患者接受了5至10年的酶替代疗法(ERT)。我们发现这些家族中在靶器官和疾病严重程度方面存在广泛的家族内表型变异性。总体而言,我们的研究结果证实了文献中先前的数据,即携带相同突变的患者存在高度的家族内表型变异性。此外,我们的结果强调了在遗传咨询期间向患者提供准确预后信息的困难,无论是在疾病进展速度还是不同器官受累方面,当这种预后仅基于患者的家族病史时。

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