Department of Psychiatry and Behavioral Sciences, University of Washington School of Medicine, Seattle.
Center for Child Health, Behavior, and Development, Seattle Children's Research Institute, WA.
Am J Speech Lang Pathol. 2019 Nov 19;28(4):1571-1581. doi: 10.1044/2019_AJSLP-19-0089. Epub 2019 Oct 3.
Purpose Youth with craniofacial microsomia (CFM) have anomalies and comorbidities that increase their risk for speech, language, and communication deficits. We examined these outcomes in youth with and without CFM and explored differences as a function of CFM phenotype and hearing status. Method Participants included youth ages 11-17 years with CFM ( = 107) and demographically similar controls ( = 306). We assessed speech intelligibility, articulation, receptive and expressive language, and parent and teacher report measures of communication. Hearing status was also screened at the study visit. Group differences were estimated using linear regression analyses with standardized effect sizes (ES) adjusted for demographic characteristics (adjusted ES) or negative binomial regression. Results Youth with CFM scored lower than unaffected peers on most measures of intelligibility, articulation, expressive language, and parent- and-teacher-rated communication. Differences were most pronounced among participants with CFM who had mandibular hypoplasia plus microtia (adjusted ES = -1.15 to -0.18). Group differences were larger in youth with CFM who failed the hearing screen (adjusted ES = -0.73 to 0.07) than in those who passed the hearing screen (adjusted ES = -0.34 to 0.27). Conclusions Youth with CFM, particularly those with mandibular hypoplasia plus microtia and/or hearing loss, should be closely monitored for speech and language concerns. Further research is needed to identify the specific needs of youth with CFM as well as to document the course of speech and language development in children with CFM.
目的 颅面短小畸形(CFM)患儿存在多种畸形和合并症,增加了其言语、语言和沟通障碍的风险。本研究旨在评估 CFM 患儿与非 CFM 对照组儿童的这些结局,并探讨 CFM 表型和听力状态的差异。
方法 参与者包括年龄在 11-17 岁的 CFM 患儿(n=107)和年龄匹配的对照组儿童(n=306)。我们评估了患儿的言语清晰度、构音、接受性和表达性语言以及家长和教师报告的沟通情况。在研究访视时还对听力状况进行了筛查。采用线性回归分析估计组间差异,采用标准化效应量(ES)调整人口统计学特征(调整后 ES)或负二项回归分析。
结果 CFM 患儿在言语清晰度、构音、表达性语言以及家长和教师评定的沟通方面的得分均显著低于对照组儿童。在伴有下颌骨发育不全和小耳畸形的 CFM 患儿中,差异最为明显(调整后 ES=-1.15 至-0.18)。在未通过听力筛查的 CFM 患儿中,组间差异较大(调整后 ES=-0.73 至 0.07),而在通过听力筛查的患儿中,组间差异较小(调整后 ES=-0.34 至 0.27)。
结论 CFM 患儿,尤其是伴有下颌骨发育不全和小耳畸形和/或听力损失的患儿,应密切关注其言语和语言问题。需要进一步研究以确定 CFM 患儿的具体需求,并记录 CFM 患儿言语语言发育的过程。