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一名患有2型神经纤维瘤病并伴有脑膜血管瘤病且累及白质的青少年死于浴缸中。

Death in a bathtub of an adolescent with neurofibromatosis type 2 exhibiting meningioangiomatosis with white matter involvement.

作者信息

Sakai Kentaro, Hayashi Kino

机构信息

Tokyo Medical Examiner's Office, Tokyo Metropolitan Government, 4-21-18 Otsuka, Bunkyo-ku, Tokyo, 112-0012, Japan.

Department of Forensic Medicine, Juntendo University School of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 113-8421, Japan.

出版信息

Forensic Sci Med Pathol. 2025 Mar;21(1):283-289. doi: 10.1007/s12024-024-00867-8. Epub 2024 Aug 24.

Abstract

Neurofibromatosis type 2 (NF2) is a neurocutaneous syndrome characterized by the development of multiple benign tumors, including vestibular schwannomas and meningiomas, in the nervous system. Seizures are rarely associated with NF2, and the lethality of this condition typically stems from tumor growth and related complications, leaving the incidence of sudden death largely unreported. This report discribes a 16-year-old girl with a history of NF2 and occasional seizures who died unexpectedly in a bathtub. Postmortem examination revealed multiple tumors in the cranial nerves (schwannoma), under the dura mater (meningioma), and in the upper cervical cord (neurofibroma). Typical signs of drowning, such as foam in the airways, were not present. Upon histological examination, meningioangiomatosis (MA) was observed in the cerebellum and the cerebral cortex, specifically in the frontal lobe, temporal lobe, and insula. The MA extended into the white matter, exhibiting severe perivascular fibrosis and cystic dilatation of perivascular spaces in the frontal lobe and cerebellum. Additionally, glial microhamartomas were detected both around and separate from the MA. These autopsy findings suggest that sudden unexpected death in epilepsy (SUDEP) was the cause of death rather than drowning. Moreover, while NF2-associated MA is typically asymptomatic, unlike sporadic MA, which commonly presents with seizures, the spread of MA into the white matter is unusual in an NF2 patient. Therefore, MA with the white matter involvement could have been a factor causing the seizures and the occurrence of SUDEP in this NF2 patient.

摘要

2型神经纤维瘤病(NF2)是一种神经皮肤综合征,其特征是在神经系统中形成多个良性肿瘤,包括前庭神经鞘瘤和脑膜瘤。癫痫发作很少与NF2相关,这种疾病的致死率通常源于肿瘤生长及相关并发症,因此猝死的发生率基本上没有报道。本报告描述了一名16岁有NF2病史且偶发癫痫的女孩,她在浴缸中意外死亡。尸检发现颅神经(神经鞘瘤)、硬脑膜下(脑膜瘤)和颈上段脊髓(神经纤维瘤)有多个肿瘤。气道中没有典型的溺水迹象,如泡沫。组织学检查发现,小脑和大脑皮层,特别是额叶、颞叶和岛叶有脑膜血管外皮细胞瘤(MA)。MA延伸至白质,额叶和小脑出现严重的血管周围纤维化和血管周围间隙的囊性扩张。此外,在MA周围及与MA分离处均检测到胶质微错构瘤。这些尸检结果表明,癫痫性猝死(SUDEP)是死因,而非溺水。此外,与通常无症状的NF2相关MA不同,散发性MA通常伴有癫痫发作,MA在NF2患者中向白质扩散并不常见。因此,MA累及白质可能是该NF2患者癫痫发作及发生SUDEP的一个因素。

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