Lay-Son Guillermo, Palomares Mirta, Guzman M Luisa, Vasquez Marcos, Puga Alonso, Repetto Gabriela M
Center for Human Genetics, Facultad de Medicina, Clínica Alemana-Universidad del Desarrollo, Chile.
Int J Pediatr Otorhinolaryngol. 2012 Dec;76(12):1726-8. doi: 10.1016/j.ijporl.2012.08.010. Epub 2012 Aug 29.
Chromosome 22q11 microdeletion syndrome (del22q11) is the most frequent microdeletion syndrome in humans, with an estimated incidence of 1/4000. It is recognized as a common identifiable cause of cleft palate. We characterized palatal abnormalities in a large cohort of Chilean patients with del22q11.
Patients with the deletion were evaluated by geneticists and speech pathologists, including nasopharyngoscopy when indicated. Comparisons between groups with and without palatal abnormalities were performed using Fisher's exact test and Mann-Whitney U test.
Two hundred and one patients were included in the study. Palate abnormalities were present in 154 patients (76.6%). The most frequent finding was submucous cleft palate (both classic and occult forms) seen in 80 patients (39.8% of the total group). Overt cleft palate or cleft lip/palate was seen in 30 patients (14.9%). Patients without palate abnormalities had significantly greater frequency of congenital heart disease and higher mortality.
Our data show a high frequency of palate abnormalities without significant association with congenital heart disease. The most common types of palate defects seen in this series are usually not evident on physical examination and thus require a high index of suspicion and active evaluation through nasopharyngoscopy.
22q11染色体微缺失综合征(del22q11)是人类最常见的微缺失综合征,估计发病率为1/4000。它被认为是腭裂的常见可识别病因。我们对一大群患有del22q11的智利患者的腭部异常进行了特征描述。
由遗传学家和言语病理学家对缺失患者进行评估,必要时包括鼻咽镜检查。使用Fisher精确检验和Mann-Whitney U检验对有和没有腭部异常的组进行比较。
201名患者纳入研究。154名患者(76.6%)存在腭部异常。最常见的发现是80名患者(占总组的39.8%)出现黏膜下腭裂(经典型和隐匿型)。30名患者(14.9%)出现明显腭裂或唇腭裂。没有腭部异常的患者先天性心脏病发生率显著更高且死亡率更高。
我们的数据显示腭部异常发生率高,与先天性心脏病无显著关联。本系列中所见的最常见腭裂类型通常在体格检查中不明显,因此需要高度怀疑并通过鼻咽镜检查进行积极评估。