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脑颅皮肤脂肪瘤病:哈伯兰德综合征

Encephalocraniocutaneous Lipomatosis: Haberland Syndrome.

作者信息

Özdoğan Selçuk, Saymaz Ceyhun, Yaltırık Cumhur Kaan, Düzkalır Hanife Gülden, Kaya Mustafa, Demirel Nail, Düzkalır Ali Haluk, Sarıkaya Başar, Aktekin Berrin

机构信息

Department of Neurosurgery, Istanbul Training and Research Hospital, Istanbul, Turkey.

Department of Neurology, Haydarpaşa Training and Research Hospital, Istanbul, Turkey.

出版信息

Am J Case Rep. 2017 Dec 1;18:1271-1275. doi: 10.12659/ajcr.907685.

DOI:10.12659/ajcr.907685
PMID:29192135
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5723105/
Abstract

BACKGROUND Encephalocraniocutaneous lipomatosis (ECCL) was first announced as a new type of ectomesodermal dysgenesis in 1970 by Haberland and Perou. ECCL was first described in 1970, and approximately 60 cases have been reported since then. The classic triad of ECCL are skin, ocular, and central nervous system involvement, including conditions such as unilateral porencephalic cyst, ipsilateral lipomatous hamartoma of the scalp-eyelids-eye globe, cortical atrophy, cranial asymmetry, developmental delay, seizures, mental retardation, and spasticity of the contralateral limbs. The dermatological hallmark is a hairless fatty tissue nevus of the scalp called nevus psiloliparus. CASE REPORT An 11-year-old right-handed boy, born at full term, was referred to our clinic. His family had no consanguinity or history of neurocutaneous disease. The patient's physical examination revealed a large hairless lesion on the right frontoparietal scalp called nevus psiloliparus. Beginning from the birth, a dermolipoma (an uncommon benign tumor) was reported to have occurred on the conjunctiva, mostly ipsilateral in his right eye and present on the ipsilateral side of the neurological abnormalities shown on magnetic resonance imaging and computed tomography. The patient had muscle weakness in left upper and lower extremities. He had a mild form of mental retardation. CONCLUSIONS There is no specific treatment for ECCL. Management of ECCL is usually symptomatic. Surgical correction of a cutaneous lesion can be performed for cosmetic improvement. An early diagnosis of ECCL allows for early symptom treatment and improved patient quality of life.

摘要

背景

1970年,哈伯兰德和佩鲁首次宣布脑颅皮肤脂肪瘤病(ECCL)是一种新型的外胚层间充质发育异常。ECCL于1970年首次被描述,自那时起已报告了约60例病例。ECCL的典型三联征包括皮肤、眼部和中枢神经系统受累,如单侧脑穿通性囊肿、同侧头皮 - 眼睑 - 眼球的脂肪瘤性错构瘤、皮质萎缩、颅骨不对称、发育迟缓、癫痫发作、智力障碍以及对侧肢体痉挛。皮肤病学特征是头皮上一种无毛的脂肪组织痣,称为无毛脂痣。

病例报告

一名11岁的右利手男孩,足月出生,被转诊至我们的诊所。他的家族无近亲结婚史或神经皮肤疾病史。患者体格检查发现右额顶部头皮有一个大的无毛病变,即无毛脂痣。自出生起,据报告其结膜上出现了一个皮样脂肪瘤(一种罕见的良性肿瘤),大多在右眼同侧,且位于磁共振成像和计算机断层扫描显示的神经异常同侧。患者左上肢和下肢肌肉无力。他有轻度智力障碍。

结论

ECCL没有特效治疗方法。ECCL的治疗通常是对症治疗。为改善美观可对皮肤病变进行手术矫正。ECCL的早期诊断有助于早期症状治疗并提高患者生活质量。

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