Natural history of brain arteriovenous malformations in hereditary hemorrhagic telangiectasia: a longitudinal neuroimaging study and review of the literature.

Boyi Li, Karen Smith, Brayan Corona-Macedo, Bilal Battal, Raj Kasthuri, Sheng-Che Hung

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Article in Neuroradiology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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6 authors.

Boyi LiDepartment of Neurosurgery, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA. boyi.li@unchealth.unc.edu.ORCID https://orcid.org/0000-0001-9516-7054

Karen SmithDepartment of Hematology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.

Brayan Corona-MacedoSchool of Medicine, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.

Bilal BattalDepartment of Radiology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.

Raj KasthuriDepartment of Hematology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.

Sheng-Che HungDepartment of Radiology, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.ORCID https://orcid.org/0000-0001-6288-3556

Funding

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6 · The paper itself

Abstract

purposeThe natural history of brain arteriovenous malformations (bAVMs) in Hereditary Hemorrhagic Telangiectasia (HHT) remains poorly understood and it has been suggested that they may have a benign course different from sporadic bAVMs. Here, we present a single-center longitudinal study evaluating the growth of bAVMs over time as assessed by serial MRIs.

methodsWe performed a retrospective analysis of 44 patients with suspected bAVMs followed at an HHT Center of Excellence from 2014 to 2024. We excluded patients that did not have serial MRIs and/or poor-quality MRIs, and patients without radiographic bAVMs. Primary outcome was growth of bAVM as measured by largest dimension on T1 post-contrasted weighted sequences of serial MRIs. Secondary outcome was hemorrhage from bAVM rupture.

resultsTwelve patients (3 male, 9 female) met our criteria. Average age at first MRI was 39.9 years (range 6-65 years). Ten patients had micro-bAVMs and 3/10 of these patients had multiple micro-bAVMs. None of the micro-bAVMs showed any growth on serial MRIs. The other two patients in our study had bAVMs with niduses > 1 cm and showed an average rate of growth of 1.61 mm/year. Ultimately, none of our patients had rupture of bAVMs over an average radiographic follow-up of 4.15 years and a total of 49.78 patient years.

conclusionOur study suggests that bAVMs in HHT patients may have a benign natural history. We found no hemorrhages and noted minimal growth of bAVMs on serial MRIs. Our results suggest HHT patients with bAVMs may not warrant frequent surveillance imaging.

Indexed as

Arteriovenous malformationBrainHereditary hemorrhagic telangiectasiaMagnetic resonance imaging

Identifiers

PMID42825815

DOI10.1186/s13023-024-03402-8

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