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病例报告:1例患有哈伯兰德综合征患者的胶质母细胞瘤病例

Case Report: A Case of Glioblastoma in a Patient With Haberland Syndrome.

作者信息

Ferranti Silvia, Sardi Iacopo, Guidi Milena, Lembo Chiara, Grosso Salvatore

机构信息

Department of Molecular Medicine and Development, University of Siena, Siena, Italy.

Department of Pediatric Oncology, Meyer Children's Hospital, Florence, Italy.

出版信息

Front Pediatr. 2021 Mar 8;9:648717. doi: 10.3389/fped.2021.648717. eCollection 2021.

Abstract

Haberland syndrome or encephalocraniocutaneous lipomatosis is a rare ectomesodermal dysgenesis defined by the triad including ocular, skin, and central nervous system involvement, which is commonly unilateral. This disorder is attributed to a post-zygotic mutation responsible for a neural tube and neural crest dysgenesis. We report the case of a 15-year-old female with Haberland syndrome with pharmacoresistant epilepsy who developed a World Health Organization-grade IV glioblastoma. This is the first case of pediatric glioblastoma associated with Haberland syndrome. The previously reported pediatric cases included benign brain tumors. To our knowledge, this is the fifth case of brain tumor associated with encephalocraniocutaneous lipomatosis and the second case of glioblastoma associated with this syndrome. The hypothesis that Haberland syndrome is associated with an increased risk of tumor development is intriguing, although the rarity of the condition is nowadays preventing us from drawing definitive conclusions about this potential link between the two entities. Further studies are needed to establish the real relationship between encephalocraniocutaneous lipomatosis and the risk of brain tumors.

摘要

哈伯兰综合征或脑颅皮肤脂肪瘤病是一种罕见的外胚层间充质发育异常,其特征为三联征,包括眼部、皮肤和中枢神经系统受累,通常为单侧性。这种疾病归因于导致神经管和神经嵴发育异常的合子后突变。我们报告了一例15岁患有哈伯兰综合征且药物难治性癫痫的女性患者,该患者发生了世界卫生组织IV级胶质母细胞瘤。这是首例与哈伯兰综合征相关的儿童胶质母细胞瘤。先前报道的儿童病例包括良性脑肿瘤。据我们所知,这是第五例与脑颅皮肤脂肪瘤病相关的脑肿瘤病例,也是第二例与该综合征相关的胶质母细胞瘤病例。哈伯兰综合征与肿瘤发生风险增加相关的假说很有趣,尽管该疾病罕见,目前我们无法就这两个实体之间的潜在联系得出明确结论。需要进一步研究以确定脑颅皮肤脂肪瘤病与脑肿瘤风险之间的真实关系。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5d36/7982648/67d6bf28a17f/fped-09-648717-g0001.jpg

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