炎症性肠病患儿的脑静脉血栓形成：临床病例

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炎症性肠病可并发中枢及外周静脉血栓形成，而在儿童期最具生命危险的累及部位为脑血管。脑静脉血栓形成约发生于3%的炎症性肠病患儿，且最常见于疾病活动期。本文报道4例脑静脉血栓形成病例，其中3例发生于溃疡性结肠炎患儿，1例发生于克罗恩病患者。第一例为8岁男童，既往有新生儿期脑缺血史，6岁时确诊溃疡性结肠炎。该病呈慢性复发性病程，在一次急性加重时并发肠道出血，因而被迫行急诊结肠切除术并造回肠造口。术后1.5个月发生急性脑循环障碍（缺血性脑卒中）。目前患儿仍遗留痉挛性偏瘫。第二例为10岁男童，溃疡性结肠炎呈慢性持续性病程，对激素治疗及生物制剂治疗均耐药，最终因 脑动脉血栓形成所致脑梗死而死亡。第三例为6岁女童，属极早发型炎症性肠病（2岁确诊为溃疡性结肠炎），在维持治疗下缓解持续3年，于一次复发期间发生 上矢状窦急性血栓形成。第四例为16岁青少年，克罗恩病并发回肠狭窄、肠间瘘及肛周瘘管，在激素治疗开始1周后 发生 上矢状窦血栓形成。该危及生命的并发症影响了患者的外科治疗策略，迫使最初先行造口手术，而非根治性手术。本组患者发生脑静脉血栓形成的危险因素包括：疾病早发、持续性病程、遗传易感性、反复激素治疗、重度活动期及外科手术干预。通过个体化治疗方案及合理的治疗策略，多数患儿可获得积极的治疗效果。对于炎症性肠病患儿，尤其是在重度活动期及合并静脉血栓高危因素时，应高度重视并及时鉴别血管并发症。脑静脉血栓形成患儿需接受动态随访及多学科联合管理，必须有神经外科医师、神经科医师及康复专科医师参与。遗憾的是，实践中医师对炎症性肠病相关并发症缺乏足够认识，导致诊断延迟，甚至出现致死结局。

作者简介

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 代码: 7773-3520

MD, Dr. Sci. (Medicine)

俄罗斯联邦, Moscow

Olga V. Shcherbakova

Federal Scientific and Clinical Center for Children and Adolescents of the Federal Medical and Biological Agency of Russia

编辑信件的主要联系方式.
Email: olga-03@yandex.ru
ORCID iD: 0000-0002-8514-3080
SPIN 代码: 3478-8606

MD, Dr. Sci. (Medicine)

俄罗斯联邦, Moscow

Saidkhasan 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 代码: 1247-1019

MD, Dr. Sci. (Medicine)

俄罗斯联邦, Moscow

Ilya 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 代码: 6329-1260

MD, Dr. Sci. (Medicine)

俄罗斯联邦, Moscow

参考

  1. 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
  2. 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
  3. 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
  4. 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
  5. 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
  6. 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
  7. 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
  8. 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
  9. 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
  10. 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
  11. 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
  12. Kalbag RM, Wolf AL. Cerebral venous thrombosis. London: Oxford University Press; 1967. P. 2–146.
  13. 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
  14. 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
  15. 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
  16. 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
  17. 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
  18. 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
  19. 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
  20. 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
  21. 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.
  22. 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.
  23. 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
  24. 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
  25. 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
  26. 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
  27. 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
  28. 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
  29. 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
  30. 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
  31. 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
  32. 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
  33. 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
  34. 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
  35. 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
  36. 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
  37. 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
  38. 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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