Hemangioblastoma and arteriovenous malformation in the same patient: a not random association or two isolated entities? Systematic review starting from a unique case

IF 1.5 4区医学 Q4 CLINICAL NEUROLOGY

Neurochirurgie Pub Date : 2024-02-06 DOI:10.1016/j.neuchi.2024.101537

Anna Maria Auricchio , Francesco Calvanese , Anni Pohjola , Aki Laakso , Mika Niemelä

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引用次数: 0

Abstract

Background

The association between intracranial hemangioblastomas and arteriovenous malformations has been documented in very few cases in literature since 1965 and might present in three modalities: "intermixed, adjacent and separated (spatially and temporally)”. Often, the pattern of presentation is “intermixed”. According to our systematic review, we propose an adjustment of the previous classification, specifically for these entities. We describe the first case of a truly “spatially separated” association between these two lesions.

Methods

Our study encompassed all adult patients diagnosed with both intracranial hemangioblastoma and AVM who were evaluated in the last 20-year period, from 2003 to 2023 at Helsinki University Hospital. Cases of this coexistence were retrospectively identified and collected from clinical records. For the systematic review, studies reporting the coexistence of hemangioblastoma and AVM in adult patients (>18 years old) were selected. Given the rarity of this pattern, case reports were also included.

Results

The combined analysis of our systematic review and institutional retrospective study revealed a total of only seven identified cases. We applied the classification of neoplasms and AVM by Yano, modifying and adapting it into our screened patient series. We systematically reclassified “adjacent” and genuinely “spatially separated” patterns based on the vascular axis supplying both lesions.

Conclusions

Hemangioblastomas and AVMs rarely coexist in the same patient. Our study reports the first instance of a truly "spatially separated" sporadic association between these vascular lesions. The rarity of such coexistence underscores the need for a nuanced and systematic classification to guide the management of these infrequent cases.

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同一患者的血管母细胞瘤和动静脉畸形：非随机关联还是两个孤立的实体？从一个独特病例开始的系统回顾

背景自 1965 年以来，颅内血管母细胞瘤与动静脉畸形之间的关联在文献中仅有极少数病例被记录在案：有三种表现形式："混合型、相邻型和分离型（空间和时间上）"。通常的表现模式是 "混合型"。根据我们的系统回顾，我们建议调整以前的分类，专门针对这些实体。我们的研究涵盖了赫尔辛基大学医院在过去 20 年（2003 年至 2023 年）中所有被诊断出同时患有颅内血管母细胞瘤和动静脉畸形的成年患者。这种并存病例是通过回顾性方法从临床记录中发现和收集的。系统综述选择了报告成年患者（18 岁）同时患有血管母细胞瘤和动静脉畸形的研究。结果对我们的系统综述和机构回顾性研究进行综合分析后发现，总共只有 7 例确定的病例。我们采用了矢野的肿瘤和动静脉畸形分类法，并对其进行了修改和调整，以适应我们筛选出的患者系列。根据供应两个病变的血管轴，我们对 "相邻 "和真正的 "空间分离 "模式进行了系统的重新分类。我们的研究首次报告了这些血管病变之间真正 "空间上分离 "的散发性关联。这种并存的罕见性突出表明，需要一种细致而系统的分类方法来指导这些罕见病例的治疗。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Neurochirurgie 医学-临床神经学

CiteScore

2.70

自引率

6.20%

发文量

100

审稿时长

29 days

期刊介绍： Neurochirurgie publishes articles on treatment, teaching and research, neurosurgery training and the professional aspects of our discipline, and also the history and progress of neurosurgery. It focuses on pathologies of the head, spine and central and peripheral nervous systems and their vascularization. All aspects of the specialty are dealt with: trauma, tumor, degenerative disease, infection, vascular pathology, and radiosurgery, and pediatrics. Transversal studies are also welcome: neuroanatomy, neurophysiology, neurology, neuropediatrics, psychiatry, neuropsychology, physical medicine and neurologic rehabilitation, neuro-anesthesia, neurologic intensive care, neuroradiology, functional exploration, neuropathology, neuro-ophthalmology, otoneurology, maxillofacial surgery, neuro-endocrinology and spine surgery. Technical and methodological aspects are also taken onboard: diagnostic and therapeutic techniques, methods for assessing results, epidemiology, surgical, interventional and radiological techniques, simulations and pathophysiological hypotheses, and educational tools. The editorial board may refuse submissions that fail to meet the journal''s aims and scope; such studies will not be peer-reviewed, and the editor in chief will promptly inform the corresponding author, so as not to delay submission to a more suitable journal. With a view to attracting an international audience of both readers and writers, Neurochirurgie especially welcomes articles in English, and gives priority to original studies. Other kinds of article - reviews, case reports, technical notes and meta-analyses - are equally published. Every year, a special edition is dedicated to the topic selected by the French Society of Neurosurgery for its annual report.