Strömland Kerstin, Miller Marilyn, Sjögreen Lotta, Johansson Maria, Joelsson Britt-Marie Ekman, Billstedt Eva, Gillberg Christopher, Danielsson Susanna, Jacobsson Catharina, Andersson-Norinder Jan, Granström Gösta
Department of Ophthalmology, The Sahlgrenska Academy at Göteborg University, Göteborg, Sweden.
Am J Med Genet A. 2007 Jun 15;143A(12):1317-25. doi: 10.1002/ajmg.a.31769.
Swedish patients with the oculo-auriculo-vertebral (OAV) spectrum participated in a prospective multidisciplinary investigation. The aims of the study were to describe their systemic and functional defects, especially autism spectrum disorders, and to search for possible etiologic risk factors. Available medical records were studied and the mothers answered a questionnaire on history of prenatal events. A clinical examination evaluating systemic findings, vision, hearing, speech, oral and swallowing function, and neuropsychiatric function, especially autism, was made. Eighteen patients, (11 males, 7 females) aged 8 months to 17 years with OAV were studied. Most frequent systemic malformations included, ear abnormalities (100%), ocular malformations (72%), vertebral deformities (67%), cerebral anomalies (50%), and congenital heart defects (33%). Functional defects consisted of hearing impairment (83%), visual impairment (28%), both visual and hearing impairment (28%), difficulties in feeding/eating (50%), speech (53%), mental retardation (39%), and severe autistic symptoms (11%). Three children were born following assisted fertilization (two intracytoplasmatic sperm injection, one in vitro fertilization), two mothers reported early bleedings, and six (33%) mothers had smoked during pregnancy.
患有眼-耳-脊椎(OAV)谱系疾病的瑞典患者参与了一项前瞻性多学科调查。该研究的目的是描述他们的全身和功能缺陷,尤其是自闭症谱系障碍,并寻找可能的病因风险因素。研究了现有的病历,母亲们回答了一份关于产前事件史的问卷。进行了一项临床检查,评估全身检查结果、视力、听力、言语、口腔和吞咽功能以及神经精神功能,尤其是自闭症。对18名年龄在8个月至17岁的OAV患者(11名男性,7名女性)进行了研究。最常见的全身畸形包括耳部异常(100%)、眼部畸形(72%)、脊柱畸形(67%)、脑部异常(50%)和先天性心脏缺陷(33%)。功能缺陷包括听力障碍(83%)、视力障碍(28%)、视力和听力障碍(28%)、喂养/进食困难(50%)、言语障碍(53%)、智力迟钝(39%)和严重自闭症症状(11%)。三名儿童通过辅助受精出生(两名胞浆内单精子注射,一名体外受精),两名母亲报告有早期出血,六名(33%)母亲在怀孕期间吸烟。