Cerebral venous thrombosis in children with inflammatory bowel disease: case reports
- Authors: Alieva E.I.1, Shcherbakova O.V.1, Bataev S.M.1, Zyabkin I.V.1
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Affiliations:
- Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia
- Issue: Vol 15, No 4 (2025)
- Pages: 557-570
- Section: Case reports
- URL: https://ogarev-online.ru/2219-4061/article/view/380594
- DOI: https://doi.org/10.17816/psaic1941
- EDN: https://elibrary.ru/ALUTIU
- ID: 380594
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Abstract
Inflammatory bowel disease may be complicated by central and peripheral venous thrombosis, with cerebral vessel involvement representing the most life-threatening localization in childhood. Cerebral venous thrombosis occurs in approximately 3% of children with inflammatory bowel disease and most frequently develops during disease exacerbation. This article presents four cases of cerebral venous thrombosis: three in children with ulcerative colitis and one in a patient with Crohn disease. The first case describes an 8-year-old boy with ulcerative colitis, a history of neonatal cerebral ischemia, and intestinal disease onset at 6 years of age. Chronic relapsing ulcerative colitis was complicated during an acute exacerbation by intestinal bleeding, requiring emergency colectomy with ileostomy. One and a half months after surgery, the patient developed an acute cerebrovascular event (ischemic stroke). To date, the child has persistent spastic hemiparesis. The second case involves a 10-year-old boy with chronic continuous ulcerative colitis refractory to both glucocorticoid and biologic therapy, who died following a cerebral infarction caused by thrombosis of the cerebral arteries. The third case is a very-early-onset inflammatory bowel disease in a 6-year-old girl (ulcerative colitis diagnosed at 2 years of age), who had maintained remission for 3 years on maintenance therapy; during a disease exacerbation, she developed acute thrombosis of the superior sagittal sinus. The fourth case describes a 16-year-old adolescent with Crohn disease complicated by ileal strictures and enteroenteric and pararectal fistulas; sagittal sinus thrombosis developed one week after initiation of glucocorticoid therapy. This life-threatening condition influenced surgical strategy, necessitating an initial diverting stoma rather than immediate radical surgery. Risk factors for cerebral venous thrombosis in our patients included early disease onset, continuous disease course, genetic predisposition, multiple courses of steroid therapy, severe disease activity, and surgical treatment. An individualized approach and tailored treatment strategy allow favorable outcomes to be achieved in most cases. In children with inflammatory bowel disease — especially during severe disease activity and in the presence of thrombotic risk factors — early identification and differentiation of vascular complications are critically important. Patients with cerebral venous thrombosis require follow-up and a multidisciplinary approach with mandatory involvement of neurosurgeons, neurologists, and rehabilitation specialists. Unfortunately, practicing physicians remain insufficiently aware of these complications of inflammatory bowel disease, which contributes to delayed diagnosis and fatal outcomes.
About the authors
Elmira I. Alieva
Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia
Email: el-alieva@yandex.ru
ORCID iD: 0009-0001-5863-1556
SPIN-code: 7773-3520
MD, Dr. Sci. (Medicine)
Russian Federation, MoscowOlga V. Shcherbakova
Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia
Author for correspondence.
Email: olga-03@yandex.ru
ORCID iD: 0000-0002-8514-3080
SPIN-code: 3478-8606
MD, Dr. Sci. (Medicine)
Russian Federation, MoscowSaidkhasan M. Bataev
Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia
Email: khassan-2@yandex.ru
ORCID iD: 0000-0003-0191-1116
SPIN-code: 1247-1019
MD, Dr. Sci. (Medicine)
Russian Federation, MoscowIlya V. Zyabkin
Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia
Email: ZyabkinIV@kidsfmba.ru
ORCID iD: 0000-0002-9717-5872
SPIN-code: 6329-1260
MD, Dr. Sci. (Medicine)
Russian Federation, MoscowReferences
- Lazzerini M, Bramuzzo M, Maschio M, et al. Thromboembolism in pediatric inflammatory bowel disease: systematic review. Inflamm Bowel Dis. 2011;17(10):2174–2183. doi: 10.1002/ibd.21563
- Gala D, Newsome T, Roberson N, et al. Thromboembolic events in patients with inflammatory bowel disease: A comprehensive overview. Diseases. 2022;10(4):73. doi: 10.3390/diseases10040073
- Lloyd-Still JD, Tomasi L. Neurovascular and thromboembolic complications of inflammatory bowel disease in childhood. J Pediatr Gastroenterol Nutr. 1989;9(4):461–466. doi: 10.1097/00005176-198911000-00012
- Standridge S, de los Reyes E. Inflammatory bowel disease and cerebrovascular arterial and venous thromboembolic events in 4 pediatric patients: a case series and review of the literature. J Child Neurol. 2008;23(1):59–66. doi: 10.1177/0883073807308706
- Saubermann LJ, Deneau M, Falcone RA, et al. Hepatic issues and complications associated with inflammatory bowel disease: A clinical report from the NASPGHAN inflammatory bowel disease and hepatology committees. J Pediatr Gastroenterol Nutr. 2017;64(4):639–652. doi: 10.1097/MPG.0000000000001492
- Nylund CM, Goudie A, Garza JM, et al. Venous thrombotic events in hospitalized children and adolescents with inflammatory bowel disease. J Pediatr Gastroenterol Nutr. 2013;56(5):485–491. doi: 10.1097/MPG.0b013e3182801e43
- Rivera-Suazo Y, Argüello Calderon I, Vázquez-Frias R. Cerebral venous sinus thrombosis in a pediatric patient with inflammatory bowel disease: A case report. Rev Gastroenterol Mex (Engl Ed). 2020;85(3):364–366. doi: 10.1016/j.rgmx.2019.07.007
- Marjot T, Yadav S, Hasan N, et al. Genes associated with adult cerebral venous thrombosis. Stroke. 2011;42(4):913–918. doi: 10.1161/STROKEAHA.110.602672
- Fong J, Rose D, Katzan I, Lopez R. Stroke in patients with inflammatory bowel disease. Inflamm Bowel Dis. 2008;14(1):S14. doi: 10.1097/00054725-200801001-00045
- Bargen JA, Barker NW. Extensive arterial and venous thrombosis complicating chronic ulcerative colitis. Arch Intern Med. 1936;58(1):17–31. doi: 10.1001/archinte.1936.00170110025002
- Harrison MJG, Truelove SC. Cerebral venous thrombosis as a complication of ulcerative colitis. Am J Dig Dis. 1967;12:1025–1028. doi: 10.1007/BF02233262
- Kalbag RM, Wolf AL. Cerebral venous thrombosis. London: Oxford University Press; 1967. P. 2–146.
- Markowitz RL, Ment LR, Gryboski JD. Cerebral thromboembolic disease in pediatric and adult inflammatory bowel disease: case report and review of the literature. J Pediatr Gastroenterol Nutr. 1989;8(3):413–420. doi: 10.1097/00005176-198904000-00028
- Calderon A, Wong JW, Becker LE. Multiple cerebral venous thromboses in a child with ulcerative colitis. Clin Pediatr (Phila). 1993;32(3):169–171. doi: 10.1177/000992289303200310
- Gormally SM, Bourke W, Kierse B, et al. Isolated cerebral thrombo-embolism and Crohn disease. Eur J Pediatr. 1995;154(10):815–818. doi: 10.1007/BF01959788
- Bridger S, Evans N, Parker A, Cairns SR. Multiple cerebral venous thromboses in a child with inflammatory bowel disease. J Pediatr Gastroenterol Nutr. 1997;25(5):533–536. doi: 10.1097/00005176-199711000-00009
- Keene DL, Matzinger MA, Jacob PJ, Humphreys P. Cerebral vascular events associated with ulcerative colitis in children. Pediatr Neurol. 2001;24(3):238–243. doi: 10.1016/s0887-8994(00)00264-2
- Kao A, Dlugos D, Hunter JV, et al. Anticoagulation therapy in cerebral sinovenous thrombosis and ulcerative colitis in children. J Child Neurol. 2002;17(7):479–482. doi: 10.1177/088307380201700702
- Diakou M, Kostadima V, Giannopoulos S, et al. Cerebral venous thrombosis in an adolescent with ulcerative colitis. Brain Dev. 2011;33(1):49–51. doi: 10.1016/j.braindev.2010.04.006
- Kutluk G, Hacıfazlıoğlu NE, Horozoğlu H, et al. Cerebral venous thrombosis associated with childhood ulcerative colitis. Turk Arch Pediatr. 2013;48:160–165. doi: 10.4274/tpa.484
- Mahmoud Reza A, Firozeh H, Houman A, Mehri NS. Pseudotumor cerebri in a case of ulcerative colitis with sagittal sinus thrombosis. Iran J Pediatr. 2013;23(1):109–112.
- Rosen I, Berkovitz D, Soudack M, et al. Cerebral vein thrombosis in a child with Crohn’s disease. Isr Med Assoc J. 2007;9(8):620–621.
- Gandhi J, Mages K, Kucine N, Chien K. Venous thromboembolism in pediatric inflammatory bowel disease: A scoping review. J Pediatr Gastroenterol Nutr. 2023;77(4):491–498. doi: 10.1097/MPG.0000000000003889
- Rohani P, Taraghikhah N, Nasehi MM, et al. Cerebrovascular events in pediatric inflammatory bowel disease: A review of published cases. Pediatr Gastroenterol Hepatol Nutr. 2022;25(3):180–193. doi: 10.5223/pghn.2022.25.3.180
- Martín-Masot R, Ortiz Pérez P, Serrano Nieto J, et al. Central venous sinus thrombosis in a boy with acute severe ulcerative colitis. Front Pediatr. 2019;7:19. doi: 10.3389/fped.2019.00019
- Salloum R, Alhalabi N, Almidani MA. A case of Syrian child with cerebral infarction as an extraintestinal manifestation of ulcerative colitis. Case Rep Gastrointest Med. 2019:5984094. doi: 10.1155/2019/5984094
- Hamid M, Ahizoune A, Berri MA. Cerebral venous thrombosis secondary to ulcerative colitis: A case report with a literature review. Radiol Case Rep. 2023;18(3):1201–1204. doi: 10.1016/j.radcr.2022.12.009
- DeFilippis EM, Barfield E, Leifer D, et al. Cerebral venous thrombosis in inflammatory bowel disease. J Dig Dis. 2015;16(2):104–108. doi: 10.1111/1751-2980.12212
- Selvitop O, Poretti A, Huisman TA, Wagner MW. Cerebral sinovenous thrombosis in a child with Crohn’s disease, otitis media, and meningitis. Neuroradiol J. 2015;28(3):274–247. doi: 10.1177/1971400915589688
- Nguyen GC, Sam J. Rising prevalence of venous thromboembolism and its impact on mortality among hospitalized inflammatory bowel disease patients. Am J Gastroenterol. 2008;103(9):2272–2280. doi: 10.1111/j.1572-0241.2008.02052.x
- Wakefield AJ, Sawyerr AM, Dhillon AP, et al. Pathogenesis of Crohn’s disease: multifocal gastrointestinal infarction. Lancet. 1989;334(8671):1057–1062. doi: 10.1016/s0140-6736(89)91078-7
- Barclay AR, Keightley JM, Horrocks I, et al. Cerebral thromboembolic events in pediatric patients with inflammatory bowel disease. Inflamm Bowel Dis. 2010;16(4):677–683. doi: 10.1002/ibd.21113
- Richard N, Leroyer A, Ley D, et al. Incidence and risk factors for thromboembolic events in pediatric-onset inflammatory bowel disease: A French population-based study. Dig Liver Dis. 2025;57(2):584–594. doi: 10.1016/j.dld.2024.09.005
- Sansotta N, Di Stasio F, Amoroso A, D’Antiga L. Cerebrovascular thrombosis in pediatric ulcerative colitis: A case report. Reports. 2025;8(1):22. doi: 10.3390/reports8010022
- Bence CM, Traynor MD Jr, Polites SF, et al. The incidence of venous thromboembolism in children following colorectal resection for inflammatory bowel disease: A multi-center study. J Pediatr Surg. 2020;55(11):2387–2392. doi: 10.1016/j.jpedsurg.2020.02.020
- Liu Y, Ren D, Zhou Q, Gao L. Cerebral sinovenous thrombosis in a child with ulcerative colitis: A case report. Medicine (Baltimore). 2020;99(2):e18649. doi: 10.1097/MD.0000000000018649
- Turner D, Ruemmele FM, Orlanski-Meyer E, et al. Management of paediatric ulcerative colitis, Part 2: acute severe colitis-an evidence-based consensus guideline from the European Crohn’s and Colitis Organization and the European Society of Paediatric Gastroenterology, Hepatology and Nutrition. J Pediatr Gastroenterol Nutr. 2018;67(2):292–310. doi: 10.1097/MPG.0000000000002036
- Torrente F, Meade S, Benchimol EI, et al. Thromboprophylaxis use in paediatric inflammatory bowel disease: An international RAND appropriateness panel. J Crohns Colitis. 2022;16(10):1609–1616. doi: 10.1093/ecco-jcc/jjac073
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