Cerebellar Hemangioblastomas in a High-risk Pregnancy: A Case Report and Review of Literature

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Abstract

Introduction

Hemangioblastomas are highly vascular benign tumors that may increase in size during pregnancy. The concurrence of cerebellar hemangioblastoma in high-risk pregnancy is extremely rare and the treatment in this situation can be challenged.

Case

Here, we report a case of a 30-year-old woman in the 33rd PW who had experienced a severe headache, dizziness, vomiting, and limb weakness. Cesarean section was performed in the 34th PW, followed by neurosurgery under multidisciplinary discussion.

Discussion

The pathological exam suggested hemangioblastomas. Finally, both the pregnancy and the fetus had a good outcome.

Conclusion

This case emphasizes that the timing of surgery should be determined according to the neurological symptoms of the pregnancy and the gestational age (GA) and condition of the fetus.

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Most cited references 17

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The 2007 WHO Classification of Tumours of the Central Nervous System

David N Louis, Hiroko Ohgaki, Otmar Wiestler … (2007)

The fourth edition of the World Health Organization (WHO) classification of tumours of the central nervous system, published in 2007, lists several new entities, including angiocentric glioma, papillary glioneuronal tumour, rosette-forming glioneuronal tumour of the fourth ventricle, papillary tumour of the pineal region, pituicytoma and spindle cell oncocytoma of the adenohypophysis. Histological variants were added if there was evidence of a different age distribution, location, genetic profile or clinical behaviour; these included pilomyxoid astrocytoma, anaplastic medulloblastoma and medulloblastoma with extensive nodularity. The WHO grading scheme and the sections on genetic profiles were updated and the rhabdoid tumour predisposition syndrome was added to the list of familial tumour syndromes typically involving the nervous system. As in the previous, 2000 edition of the WHO ‘Blue Book’, the classification is accompanied by a concise commentary on clinico-pathological characteristics of each tumour type. The 2007 WHO classification is based on the consensus of an international Working Group of 25 pathologists and geneticists, as well as contributions from more than 70 international experts overall, and is presented as the standard for the definition of brain tumours to the clinical oncology and cancer research communities world-wide.

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Management Strategy for Meningioma in Pregnancy: A Clinical Study.

A Jallu, I Kanaan, S H Kanaan (2003)

We retrospectively studied 18 pregnant women from 600 cases of meningioma treated at this Institution between 1986 and 2001. The variables evaluated included clinical presentation; radiological findings; timing and extent of surgical resection; and an overview of gestational, clinical, and surgical outcomes. Visual impairment was the chief complaint followed by headache and seizures. The tumors involved the tuberculum sella (8), sphenoid wing (4), convexity (2), parasellar (2), cerebellopontine angle (CPA) (1), and anterior falx (1). Gross total resection was achieved in 14, and subtotal resection was achieved in two patients. There were no related fetal or maternal deaths. Of seven patients with advanced or progressive visual impairment who underwent resection during pregnancy, three improved noticeably, two worsened, and two were unchanged. Five other patients with visual disturbance achieved full-term spontaneous delivery with improved vision in two and improved vision after post-confinement surgery in one. Vision remained unchanged in two. Intracranial meningioma during pregnancy challenges the skill of obstetricians and neurosurgeons to secure delivery of the baby and resection of the tumor. Advances in fetal and maternal monitoring, neuroanesthesia, and microsurgical techniques allow safe neurosurgical management of these patients, and pregnancy usually continues successfully to term. Surgical intervention has no major effect on minimal residual vision or advanced optic nerve atrophy.

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Accelerated growth of hemangioblastoma in pregnancy: the role of proangiogenic factors and upregulation of hypoxia-inducible factor (HIF) in a non-oxygen-dependent pathway.

Yosef Laviv, Joshua Wang, Matthew Anderson … (2019)

Hemangioblastomas (HBs) are benign, highly vascular tumors, often characterized by loss of function of the von Hippel-Lindau (vHL) gene. They are the most common central nervous system tumor observed in vHL syndrome. Loss of function of the vHL gene creates a "pseudo-hypoxic" state, causing overactivation of hypoxia-inducible factor (HIF) and vascular endothelial growth factor (VEGF)-related pathways. In some cases, HBs can rapidly increase in size during pregnancy to then present acutely, which most frequently occurs after the 20th gestational week. These changes in size usually occur from enlargement of the cystic component of the HB. Due to their preferred location in the posterior fossa near critical structures as well as along the spinal cord, such cases can present with severe neurological deficits, requiring urgent surgical intervention in a multidisciplinary setting. However, the reasons for this acute flare-up during pregnancy remain poorly understood, as are the reasons why this occurs in only a subset of tumors. Unveiling the etiology for this clinical scenario can affect the treatment of HBs, as it will contribute to the understanding of the pathophysiology of such a transformation from a quiescent lesion to a symptomatic one, not only in the setting of pregnancy. Identifying the correct triggers and the conditions initiating and mediating this switch will enable us to develop preventive medications which should allow us to keep the tumor in its quiescent phase. In this pathophysiological review, we investigate the association between HB growth and pregnancy based on an analysis > 40 such published cases. We suggest that the proangiogenic state of pregnancy is the leading etiology for this striking association, and to support the argument, we discuss its potential impact on HIF overexpression in a non-hypoxic manner through activation of the PI3K/Akt/mTOR pathway by proangiogenic factors. Specifically, we discuss the involvement of placental growth factor (PlGF) and its receptor vascular endothelial growth factor receptor 1 (VEGFR-1) in various pathologic processes that can lead to the formation and growth of peritumoral edema and cysts, which are the primary causes for the development of any symptoms in HB. Both PlGF and VEGFR-1 are expressed at increased levels during pregnancy, and both have been reported as part of various pathological processes, including angiogenesis and tumorigenesis. The unique feature that both do essentially not show any significant negative impact on regular physiological processes makes them attractive therapeutic targets since very little side effects are expected. Further research into the effects of anti-PlGF or anti-VEGFR-1 therapy in HB is therefore recommended.

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Author and article information

Journal

Journal ID (publisher-id): CMIR

Journal ID (nlm-ta): Curr Med Imaging

Title: Current Medical Imaging

Abbreviated Title: Curr. Med. Imaging

Publisher: Bentham Science Publishers

ISSN (Print): 1573-4056

ISSN (Electronic): 1875-6603

Publication date (Electronic): 2023

Volume: 19

Issue: 6

Pages: 654-657

Affiliations

[1 ] deptDepartment of Neurosurgery , Affiliated Hospital of Southwest Medical University , 25 Taiping Street, Lu Zhou, 646000, , China, ;

[2 ] deptDepartment of Neurosurgery , The Third Hospital of MianYang·Sichuan Mental Health Center , Sichuan, , 621000, , China;

[3 ] deptDepartment of Pathology , The Third Hospital of MianYang·Sichuan Mental Health Center , Sichuan, , 621000, , China

Author notes

[* ]Address correspondence to this author at the The Third Hospital of MianYang, 190 East section of Jian Nan Road, Mianyang, Sichuan Province 621000, China; Fax: +86 0816-2296218; E-mail: ly10012002@ 123456163.com

Article

Publisher ID: CMIR-19-6-654

DOI: 10.2174/1573405619666221017114922

PMC ID: 10262636

PubMed ID: 36263473

SO-VID: bb13e728-8b00-44d6-956f-572f086c7e33

License:

This is an Open Access article published under CC BY 4.0 https://creativecommons.org/licenses/by/4.0/legalcode

History

Date received : 10 June 2022

Date revision received : 24 August 2022

Date accepted : 15 September 2022

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Cerebellar Hemangioblastomas in a High-risk Pregnancy: A Case Report and Review of Literature

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Abstract

Introduction

Case

Discussion

Conclusion

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Most cited references 17

The 2007 WHO Classification of Tumours of the Central Nervous System

Management Strategy for Meningioma in Pregnancy: A Clinical Study.

Accelerated growth of hemangioblastoma in pregnancy: the role of proangiogenic factors and upregulation of hypoxia-inducible factor (HIF) in a non-oxygen-dependent pathway.

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