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Microvascular proliferation in arteriovenous malformation of the hand worsens during pregnancy: a case report

Authors
  • Utami, Amalia M.1, 2
  • Horbach, Sophie E.R.3
  • Meijer-Jorna, Lorine B.4
  • Waas, Ingeborg S.E.1
  • de Boer, Onno J.1
  • van der Wal, Allard C.1
  • van der Horst, Chantal M.A.M.3
  • 1 Department of Pathology
  • 2 Department of Pathology Anatomy, Faculty of Medicine, Hasanuddin University, Makassar, Indonesia
  • 3 Department of Plastic Surgery, Amsterdam University Medical Center, AMC, University of Amsterdam, Amsterdam
  • 4 Symbiant Pathology, NWZ – Noordwest Ziekenhuisgroep, Alkmaar, The Netherlands
Type
Published Article
Journal
Annals of Medicine and Surgery
Publisher
Elsevier BV
Publication Date
Apr 12, 2023
Volume
85
Issue
4
Pages
1262–1269
Identifiers
DOI: 10.1097/MS9.0000000000000507
PMID: 37113922
PMCID: PMC10129217
Source
PubMed Central
Keywords
Disciplines
  • Case Reports
License
Unknown

Abstract

Arteriovenous malformations (AVMs) are rare congenital disorders characterized by episodes of disproportionate growth that can cause pain and severe bleeding, with microvascular proliferation (MVP) associated with these episodes. Hormonal influences can also worsen the symptoms in patients with AVM. Case presentation: This case report presents a female patient with congenital vascular malformations of the left hand since birth, whose symptoms worsened during puberty and pregnancy, ultimately leading to amputation of the left hand due to unbearable pain and loss of function. Pathologic analysis revealed substantial MVP activity within the tissues of the AVM, with an expression of receptors for estrogen, growth hormone, and follicle-stimulating hormone in the vessels of the AVM, including MVP areas. Resected materials not related to pregnancy revealed chronic inflammation and fibrosis but hardly any MVP. Discussion and conclusion: These findings suggest a role for MVP in the progressive growth of AVM during pregnancy, with a potential role for hormonal influences. The case highlights the relationship between AVM symptoms and size during pregnancy and the pathological findings of MVP areas within the AVM with hormone receptor expression on proliferating vessels in resected materials.

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