Sans-Fitó Anna, Campistol-Plana Jaime, Mas-Salguero María José, Póo-Argüelles Pilar, Fernández-Alvarez Emilio
Servicio de Neurologia, Unitat Integrada Hospital Clinic-Sant Joan de Déu, Universitat de Barcelona, Spain.
J Child Neurol. 2002 Feb;17(2):132-4. doi: 10.1177/088307380201700208.
Pontocerebellar hypoplasia is an autosomal recessive syndrome with onset during the fetal period. Two subtypes of pontocerebellar hypoplasia have been described on the basis of clinical and neuropathologic criteria. Pontocerebellar hypoplasia type 2 is characterized by progressive microcephaly, early onset of extrapyramidal dyskinesia, and near absence of motor and cognitive development, without signs of either spinal or peripheral involvement. We report a clinical observation of a patient with pontocerebellar hypoplasia type 2, a 3-year-old girl with progressive microcephaly, dystonic limb movements, and absence of motor and cognitive development. Cranial magnetic resonance imaging revealed pontocerebellar hypoplasia. At the age of 2 years, she suffered a Reye-like syndrome that worsened her condition. Differential diagnosis was established with intrauterine injuries, other malformative syndromes, and neurodegenerative or neurometabolic disorders, which can be associated with cerebellar hypoplasia.
脑桥小脑发育不全是一种常染色体隐性综合征,在胎儿期发病。根据临床和神经病理学标准,脑桥小脑发育不全已被描述为两种亚型。2型脑桥小脑发育不全的特征是进行性小头畸形、锥体外系运动障碍早发、几乎没有运动和认知发育,且无脊髓或周围神经受累迹象。我们报告了1例2型脑桥小脑发育不全患者的临床观察结果,该患者为一名3岁女童,有进行性小头畸形、肢体张力障碍性运动,且无运动和认知发育。头颅磁共振成像显示脑桥小脑发育不全。2岁时,她患了类似瑞氏综合征的疾病,病情恶化。通过与可伴有小脑发育不全的子宫内损伤、其他畸形综合征以及神经退行性或神经代谢性疾病进行鉴别诊断。