Yamashita Yushiro, Kusaga Akira, Koga Yasutoshi, Nagamitsu Shin-ichiro, Matsuishi Toyojiro
Department of Pediatics and Child Health, Kurume University School of Medicine, Kurume, Japan.
Pediatr Neurol. 2004 Nov;31(5):364-6. doi: 10.1016/j.pediatrneurol.2004.05.015.
This reports describes a 12-year-old Japanese female with Noonan syndrome who had antiphospholipid syndrome and moyamoya-like vascular changes. She presented choreic movements in her face and extremities. She manifested phenotypic features of Noonan syndrome with short stature, mental retardation, and a webbed neck. Magnetic resonance angiography revealed occlusion of bilateral internal carotid arteries and moyamoya-like vascular changes around the basal ganglion region. Pimozide completely resolved the patient's choreic movements. Tests for anticardiolipin antibody and lupus anticoagulant were positive. The patient has manifested no symptoms for 2 years with pimozide, aspirin, and growth hormone treatment, without further aggravation of moyamoya-like vascular changes. This article is the first report of Noonan syndrome with antiphospholipid syndrome and moyamoya-like vascular lesions.
本报告描述了一名患有努南综合征的12岁日本女性,她同时患有抗磷脂综合征和烟雾病样血管病变。她出现了面部和四肢的舞蹈样动作。她表现出努南综合征的表型特征,如身材矮小、智力迟钝和蹼颈。磁共振血管造影显示双侧颈内动脉闭塞以及基底节区周围的烟雾病样血管病变。匹莫齐特完全消除了患者的舞蹈样动作。抗心磷脂抗体和狼疮抗凝物检测呈阳性。该患者在接受匹莫齐特、阿司匹林和生长激素治疗后2年未出现症状,烟雾病样血管病变也未进一步加重。本文是关于努南综合征合并抗磷脂综合征和烟雾病样血管病变的首例报告。