Department of Otorhinolaryngology, Radboud University Nijmegen Medical Centre, PO BOX 9101, 6500 HB Nijmegen, The Netherlands.
Hear Res. 2011 May;275(1-2):81-8. doi: 10.1016/j.heares.2010.12.007. Epub 2010 Dec 10.
The aim of the study was to report otologic and audiologic characteristics in a group of children with Turner syndrome (TS) and correlate these findings to karyotype. Additionally, we give recommendations for the otologic care of these children. Sixty children (age 1.7-21.2 years) were included in this retrospective study. Medical history and karyotypes were recorded and otologic and audiologic evaluation was performed. A history of recurrent otitis media was reported in 41/60 (68%) children and 3/60 (5%) had suffered from cholesteatoma. Audiometric data in 56 children revealed that normal hearing was only present in 33/112 (29%) ears. All other ears 79/112 (71%) were classified in five different audiometric categories for hearing loss. Hearing thresholds in general appeared to be about 10-11 dB worse in children with a monosomy 45,X or isochromosome (both have a total deletion of the short (p) arm of the X-chromosome) compared to those having a mosaicism or structural anomaly (partial deletion, or total deletion in only a few cells). Our findings support the hypothesis that hearing can be affected by loss of the p-arm of the X-chromosome. It is for the first time that a relation between hearing problems and karyotype is statistically confirmed in a large group of children with TS.
本研究旨在报告特纳综合征(TS)患儿的耳部和听力特征,并将这些发现与核型相关联。此外,我们还为这些患儿的耳部护理提供了建议。在这项回顾性研究中,共纳入了 60 名儿童(年龄 1.7-21.2 岁)。记录了他们的病史和核型,并进行了耳部和听力评估。41/60(68%)名患儿有复发性中耳炎病史,3/60(5%)名患儿患有胆脂瘤。56 名儿童的听力数据显示,仅 33/112(29%)耳的听力正常。其他 79/112(71%)耳可分为五种不同的听力损失类别。与嵌合体或结构异常(部分缺失或仅少数细胞缺失)的患儿相比,单体型 45,X 或等臂染色体(X 染色体短臂(p)完全缺失)患儿的听力阈值普遍低 10-11dB。我们的研究结果支持了以下假设:听力可能受到 X 染色体 p 臂缺失的影响。这是首次在一大群 TS 患儿中,通过统计学方法证实了听力问题与核型之间的关系。