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患有家族性偏瘫性偏头痛的意大利家庭中的遗传异质性。

Genetic heterogeneity in Italian families with familial hemiplegic migraine.

作者信息

Carrera P, Piatti M, Stenirri S, Grimaldi L M, Marchioni E, Curcio M, Righetti P G, Ferrari M, Gelfi C

机构信息

IRCCS H San Raffaele, Laboratorio Biologia Molecolare Clinica, Italy.

出版信息

Neurology. 1999 Jul 13;53(1):26-33. doi: 10.1212/wnl.53.1.26.

Abstract

OBJECTIVE

To verify linkage to chromosome 19p13, to detect mutations in the CACNA1A gene, and to correlate genetic results to their clinical phenotypes in Italian families with familial hemiplegic migraine (FHM).

BACKGROUND

FHM is an autosomal dominant disease, classified as a subtype of migraine with aura. Only a proportion of FHM patients have been associated with chromosome 19p13. Among these, four missense mutations within the CACNA1A gene in five unrelated families have been described.

METHODS

A linkage study was performed in 19 patients affected by FHM from five families by studying microsatellite markers associated with the 19p13 region. All familial and seven additional sporadic patients with FHM were analyzed to search for mutations within the CACNA1A gene by applying the double gradient-denaturant gradient electrophoresis technique.

RESULTS

Lod score values did not establish significantly linkage to chromosome 19. However, seven new genetic variants were detected: six were new polymorphisms. The seventh was a missense mutation present in family 1, and it was associated with a hemiplegic migraine phenotype without unconsciousness and cerebellar ataxia. Because this missense mutation is absent in the general population and cosegregates with the disease, it may be a pathologic mutation.

CONCLUSIONS

Genetic heterogeneity of FHM has been shown in familial and sporadic FHM patients of Italian origin. The new missense mutation-G4644T-is associated with milder clinical features compared with typical FHM.

摘要

目的

在意大利患有家族性偏瘫性偏头痛(FHM)的家族中,验证与19号染色体p13区域的连锁关系,检测CACNA1A基因中的突变,并将遗传结果与其临床表型相关联。

背景

FHM是一种常染色体显性疾病,被归类为伴有先兆的偏头痛的一种亚型。只有一部分FHM患者与19号染色体p13区域相关联。在这些患者中,已描述了五个无关家族中CACNA1A基因内的四个错义突变。

方法

通过研究与19p13区域相关的微卫星标记,对来自五个家族的19名受FHM影响的患者进行了连锁研究。通过应用双梯度-变性梯度电泳技术,对所有家族性和另外七名散发性FHM患者进行分析,以寻找CACNA1A基因内的突变。

结果

连锁分析得分值未显示与19号染色体有显著连锁。然而,检测到七个新的基因变异:六个是新的多态性。第七个是家族1中存在的一个错义突变,它与无昏迷和小脑共济失调的偏瘫性偏头痛表型相关。由于这种错义突变在普通人群中不存在且与疾病共分离,它可能是一个病理性突变。

结论

在意大利裔的家族性和散发性FHM患者中已显示出FHM的遗传异质性。与典型FHM相比,新的错义突变G4644T与较轻的临床特征相关。

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