Halayem Soumeyya, Hamza Mariem, Maazoul Faouzi, Ben Turkia Hadhemi, Touati Maissa, Tebib Neji, Mrad Ridha, Bouden Asma
Department of Child Psychiatry, Razi Hospital, Manouba, Tunisia.
Faculty of Medicine of Tunis, Tunis, Tunisia.
Am J Med Genet A. 2016 Apr;170A(4):1035-9. doi: 10.1002/ajmg.a.37518. Epub 2015 Dec 22.
Simpson-Golabi-Behmel syndrome (SGBS) is an X-linked condition characterized by pre and post natal overgrowth, facial malformations, and visceral, skeletal, and neurological anomalies. The physical characteristics of SGBS have been well documented; however there is a lack of description regarding the behavioral phenotype. We report the case of a 6-year-old boy, with confirmed deletion of 6-8 exons of the glypican-3 gene (GPC3) who presents three distinctive findings: the persistence of the craniopharyngeal canal, an immune-allergic specificity, and a scarcely behavioral phenotype consisting in the association of Austim Spectrum Disorder with accompanying mild intellectual disability and language impairments. He also fulfilled the criteria of Attention Deficit Hyperactivity Disorder and Oppositional Defiant Disorder according to DSM 5 criteria. The specificities of the case are discussed in the light of recent pathophysiological data.
辛普森-戈拉比-贝梅尔综合征(SGBS)是一种X连锁疾病,其特征为出生前后过度生长、面部畸形以及内脏、骨骼和神经异常。SGBS的身体特征已有充分记录;然而,关于其行为表型的描述却很缺乏。我们报告了一例6岁男孩的病例,该男孩经证实存在磷脂酰肌醇蛋白聚糖-3基因(GPC3)第6至8外显子缺失,呈现出三个独特的表现:颅咽管持续存在、免疫过敏特异性以及一种罕见的行为表型,即孤独症谱系障碍伴有轻度智力残疾和语言障碍。根据《精神疾病诊断与统计手册》第5版标准,他还符合注意力缺陷多动障碍和对立违抗障碍的标准。我们根据最近的病理生理数据对该病例的特异性进行了讨论。