Barbi G, Scheres J M, Schindler D, Taalman R D, Rodens K, Mehnert K, Müller M, Seyschab H
Abteilung Klinische Genetik der Universität, Würzburg, Federal Republic of Germany.
Am J Med Genet. 1991 Jul 1;40(1):44-50. doi: 10.1002/ajmg.1320400109.
We report on a microcephalic, growth-retarded newborn girl without major anomalies who has chromosome instability in lymphocytes and fibroblasts. Frequent involvement of bands 7p13, 7q34, 14q11, and 14q32 suggested the diagnosis of ataxia telangiectasia (AT) or a related disorder. Supportive evidence was radioresistant DNA synthesis in fibroblasts and radiation hypersensitivity of short-term lymphocyte cultures. Follow-up for nearly 4 years showed largely normal development, and no signs of telangiectasia, ataxia, or immunodeficiency. Serum AFP levels turned from elevated at age 5 months to normal at age 2 years. We propose that our patient belongs to the expanding category of "AT-related" genetic disorders, probably to the Nijmegen breakage syndrome.
我们报告了一名头小、生长发育迟缓的新生女婴,无重大异常,其淋巴细胞和成纤维细胞存在染色体不稳定。7p13、7q34、14q11和14q32带频繁受累提示诊断为共济失调毛细血管扩张症(AT)或相关疾病。支持性证据为成纤维细胞中的放射抗性DNA合成以及短期淋巴细胞培养的辐射超敏反应。近4年的随访显示发育基本正常,无毛细血管扩张、共济失调或免疫缺陷的迹象。血清甲胎蛋白水平从5个月大时升高转变为2岁时正常。我们认为我们的患者属于不断扩大的“AT相关”遗传疾病范畴,可能属于尼美根断裂综合征。