Pediatric Neurosurgery Service, Department of Surgery, CHU Sainte-Justine, University of Montreal, Montreal, Quebec, Canada.
Division of Child Neurology, BC Children's Hospital, University of British Columbia, Vancouver, British Columbia, Canada.
Pediatr Neurol. 2018 Jul;84:27-31. doi: 10.1016/j.pediatrneurol.2017.12.016. Epub 2018 Feb 14.
Because most cases of brain tumor-associated narcolepsy have been published in the form of case reports or small series, the clinical presentation and evolution have not been well described. We sought to better define the epidemiology, etiology, and outcome of brain tumor-related narcolepsy.
We conducted an extensive review of the literature to identify cases of narcolepsy associated with brain tumors. Only cases of brain tumors involving the hypothalamic region including the suprasellar, sellar, and thalamus were included in this study.
We report a child with possible narcolepsy in a child with a brain tumor. Through our literature review, we identified 25 additional cases of narcolepsy associated with brain tumors affecting the hypothalamic area. Most symptomatic narcolepsy cases were reported in children (70%). Half of the patients (13 of 25, 52%) developed narcolepsy after surgery, whereas 11 patients (44%) were symptomatic at the time of the tumor diagnosis. Ten patients had narcolepsy without cataplexy. Most cases were associated with craniopharyngioma (38%), adenoma (24%), and glioma (14%). Three, including our patient, experienced a complete resolution of symptoms. All patients underwent biopsy and were treated with adjuvant therapy. For patients with persistent symptoms, most (60%) improved following medical management of narcolepsy.
This study represents the largest compilation of patients with this association. Our study allows us to better understand the etiology and outcome of patients with narcolepsy-related brain tumors.
由于大多数脑瘤相关性嗜睡症病例均以病例报告或小系列形式发表,因此其临床表现和演变尚未得到充分描述。我们旨在更好地定义与脑瘤相关的嗜睡症的流行病学、病因和结局。
我们广泛查阅文献,以确定与脑瘤相关的嗜睡症病例。本研究仅纳入涉及下丘脑区域(包括鞍上、蝶鞍和丘脑)的脑瘤病例。
我们报告了一例儿童脑瘤伴可能的嗜睡症病例。通过文献复习,我们共确定了 25 例另外的与影响下丘脑区域的脑瘤相关的嗜睡症病例。大多数有症状的嗜睡症病例发生在儿童中(70%)。一半的患者(25 例中的 13 例,52%)在手术后出现嗜睡症,而 11 例(44%)在肿瘤诊断时即有症状。10 例患者表现为无猝倒的嗜睡症。大多数病例与颅咽管瘤(38%)、腺瘤(24%)和胶质瘤(14%)相关。包括我们的患者在内,有 3 例患者症状完全缓解。所有患者均接受了活检和辅助治疗。对于持续存在症状的患者,大多数(60%)在接受针对嗜睡症的药物治疗后症状得到改善。
本研究是此类相关性患者最大型的病例汇编。我们的研究使我们能够更好地理解与脑瘤相关的嗜睡症患者的病因和结局。