Advertisement
Correspondence| Volume 283, P158-159, April 2023

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
Previous ArticleStraining to void at preoperative urodynamic study as a risk factor for prolapse recurrence after surgery
Next ArticleMaternal and perinatal outcome in dengue and COVID-19 co-infected pregnancies
      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.
      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'

      Subscribe:

      Subscribe to European Journal of Obstetrics and Gynecology and Reproductive Biology
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Register: Create an account
      Institutional Access: Sign in to ScienceDirect

      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.

      ScienceDirect

      Access this article on ScienceDirect

      The content on this site is intended for healthcare professionals.


      We use cookies to help provide and enhance our service and tailor content. To update your cookie settings, please visit the Cookie Preference Center for this site.
      Copyright © 2023 Elsevier Inc. except certain content provided by third parties.


      RELX