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来自土耳其中黑海地区的一个患有肯尼迪病的大家族中的表型差异。

Phenotypic differences in a large family with Kennedy's disease from the Middle Black Sea region of Turkey.

作者信息

Karaer Hatice, Kaplan Yüksel, Kurt Semiha, Gundogdu Asli, Erdoğan Begüm, Başak Nazli A

机构信息

Department of Neurology, Gaziosmanpasa University, Tokat, Turkey.

出版信息

Amyotroph Lateral Scler. 2010;11(1-2):148-53. doi: 10.3109/17482960802445086.

Abstract

We report the clinical and electrophysiological features of a large Turkish family with genetically confirmed X-linked spinal and bulbar muscular atrophy (SBMA). Family members were identified by field work. A detailed history was obtained from each subject, and each subject received a detailed neurological examination. To confirm the CAG repeat expansion in the AR gene, genomic DNA was extracted from the peripheral blood of patients. The family consisted of 128 individuals over five generations, with two consanguineous parents, one slightly affected female, and 12 affected males with SBMA. We studied the five surviving male patients and one surviving female carrier. The age at disease onset, phenotypic features, and disease severity varied among the family members. DNA analysis was performed on five individuals, belonging to five generations of the family. Four affected males and a slightly affected female carrier were shown to carry an expanded CAG repeat in the androgen receptor gene. This family report is consistent with previous studies suggesting that SBMA may be present with a wide clinical spectrum in affected family members. Further descriptions of SBMA affected families with different ethnic backgrounds may assist in identifying possible phenotypic and genetic features of the disease.

摘要

我们报告了一个经基因确诊的X连锁脊髓和延髓肌萎缩症(SBMA)的大型土耳其家族的临床和电生理特征。通过实地走访确定了家族成员。从每位受试者处获取了详细病史,且每位受试者都接受了详细的神经学检查。为确认雄激素受体(AR)基因中的CAG重复序列扩增,从患者外周血中提取了基因组DNA。该家族由五代共128人组成,有一对近亲父母、一名症状轻微的女性以及12名患有SBMA的男性。我们研究了五名存活的男性患者和一名存活的女性携带者。家族成员之间疾病发病年龄、表型特征和疾病严重程度各不相同。对属于该家族五代的五名个体进行了DNA分析。四名患病男性和一名症状轻微的女性携带者被证明在雄激素受体基因中携带扩增的CAG重复序列。这份家族报告与之前的研究一致,表明SBMA在受影响的家族成员中可能具有广泛的临床谱。对不同种族背景的SBMA受累家族的进一步描述可能有助于确定该疾病可能的表型和基因特征。

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