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Correspondence| Volume 283, P158-159, April 2023

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Pregnancy-associated ruptured splenic arteriovenous malformation: A rare but fatal complication in hereditary hemorrhagic telangiectasia

Published:February 11, 2023DOI:https://doi.org/10.1016/j.ejogrb.2023.02.004
Pregnancy-associated ruptured splenic arteriovenous malformation: A rare but fatal complication in hereditary hemorrhagic telangiectasia
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      Hereditary hemorrhagic telangiectasia (HHT), also known as Osler-Weber-Rendu syndrome, is a rare genetic disorder characterized by abnormal blood vessel formation in skin, mucous membranes, and viscera [

      McDonald J, Stevenson DA. Hereditary Hemorrhagic Telangiectasia. 2000 Jun 26 [updated 2021 Nov 24]. In: Adam MP, Everman DB, Mirzaa GM, Pagon RA, Wallace SE, Bean LJH, Gripp KW, Amemiya A, editors. GeneReviews® [Internet]. Seattle (WA): University of Washington, Seattle; 1993–2023. PMID: 20301525.

      ]. Visceral arteriovenous malformations (AVMs) in the liver, lungs, and brain are common in HHT patients, however splenic AVMs are extremely rare [
      • Welle C.L.
      • Welch B.T.
      • Brinjikji W.
      • Ehman E.C.
      • Venkatesh S.K.
      • Johnson M.P.
      • et al.
      Abdominal manifestations of hereditary hemorrhagic telangiectasia: a series of 333 patients over 15 years.
      Abdom Radiol (NY). 2019 Jul; 44 (PMID: 30888464): 2384-2391https://doi.org/10.1007/s00261-019-01976-7
      ]. Splenic AVM rupture during pregnancy has not been described previously in the literature [
      • Delagrange L.
      • Dupuis O.
      • Fargeton A.E.
      • Bernard L.
      • Decullier E.
      • Dupuis-Girod S.
      Obstetrical and neonatal complications in hereditary haemorrhagic telangiectasia: A retrospective study.
      BJOG. 2023 Feb; 130 (Epub 2022 Oct 9 PMID: 36156839): 303-311https://doi.org/10.1111/1471-0528.17303
      ,
      • Dupuis O.
      • Delagrange L.
      • Dupuis-Girod S.
      Hereditary haemorrhagic telangiectasia and pregnancy: a review of the literature.
      Orphanet J Rare Dis. 2020 Jan 7; 15: 5https://doi.org/10.1186/s13023-019-1286-z. PMID: 31910869; PMCID: PMC6947864
      ]. In this case report, we present a 30-year-old woman with HHT-Type 2 who experienced severe splenic rupture during her second pregnancy.
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      References

      1. McDonald J, Stevenson DA. Hereditary Hemorrhagic Telangiectasia. 2000 Jun 26 [updated 2021 Nov 24]. In: Adam MP, Everman DB, Mirzaa GM, Pagon RA, Wallace SE, Bean LJH, Gripp KW, Amemiya A, editors. GeneReviews® [Internet]. Seattle (WA): University of Washington, Seattle; 1993–2023. PMID: 20301525.

        View in Article
        • Welle C.L.
        • Welch B.T.
        • Brinjikji W.
        • Ehman E.C.
        • Venkatesh S.K.
        • Johnson M.P.
        • et al.
        Abdominal manifestations of hereditary hemorrhagic telangiectasia: a series of 333 patients over 15 years.
        Abdom Radiol (NY). 2019 Jul; 44 (PMID: 30888464): 2384-2391https://doi.org/10.1007/s00261-019-01976-7
        View in Article
        • Delagrange L.
        • Dupuis O.
        • Fargeton A.E.
        • Bernard L.
        • Decullier E.
        • Dupuis-Girod S.
        Obstetrical and neonatal complications in hereditary haemorrhagic telangiectasia: A retrospective study.
        BJOG. 2023 Feb; 130 (Epub 2022 Oct 9 PMID: 36156839): 303-311https://doi.org/10.1111/1471-0528.17303
        View in Article
        • Dupuis O.
        • Delagrange L.
        • Dupuis-Girod S.
        Hereditary haemorrhagic telangiectasia and pregnancy: a review of the literature.
        Orphanet J Rare Dis. 2020 Jan 7; 15: 5https://doi.org/10.1186/s13023-019-1286-z. PMID: 31910869; PMCID: PMC6947864
        View in Article
        • Faughnan M.E.
        • Mager J.J.
        • Hetts S.W.
        • Palda V.A.
        • Lang-Robertson K.
        • Buscarini E.
        • et al.
        Second International Guidelines for the Diagnosis and Management of Hereditary Hemorrhagic Telangiectasia.
        Ann Intern Med. 2020 Dec 15; 173 (Epub 2020 Sep 8 PMID: 32894695): 989-1001https://doi.org/10.7326/M20-1443
        View in Article

      Article info

      Publication history

      Published online: February 11, 2023
      Accepted: February 10, 2023
      Received: January 18, 2023

      Identification

      DOI: https://doi.org/10.1016/j.ejogrb.2023.02.004

      Copyright

      © 2023 Elsevier B.V. All rights reserved.

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