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Eur J Pediatr Surg 2023; 33(04): 279-286
DOI: 10.1055/s-0042-1751222
Original Article

Clinical Characteristics and Management of Colorectal Vascular Malformation in Children: A Retrospective Study of 23 Cases

Yanbing Huang‡   *
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Peijun Zhuang‡   *
2   Department of Anesthesiology, Children's Hospital of Fudan University, Shanghai, China
,
Gong Chen
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Yanlei Huang
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Kuiran Dong
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Xianmin Xiao
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Shan Zheng
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
,
Song Sun
1   Department of Surgery, Children's Hospital of Fudan University, Shanghai, China
› Institutsangaben Funding This study received financial support from the National Natural Science Foundation of China (nos. 81700450), Clinical Research Plan of SHDC (no. SHDC2020CR2009A), and Shanghai Municipal Key Clinical Specialty (no. shslczdzk05703). This study was approved by the Ethics Commission of Children's Hospital of Fudan University.
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Abstract

Introduction The objective of this study is to summarize the clinical characteristics and management of rare diseases of colorectal vascular malformation (CRVM) in children.

Methods We retrospectively analyzed the clinical data of CRVM patients admitted to the Children's Hospital of Fudan University from 2004 to 2019.

Results A total of 23 cases (16 males, 7 females) were enrolled. The median age of symptom onset was 1.4 years. Hematochezia and anemia were cardinal symptoms. Fourteen patients (60.9%) were misdiagnosed as anal fissures (n = 4), internal hemorrhoids (n = 3), rectal polyps (n = 2), inflammatory bowel disease (n = 2), portal hypertension (n = 2), and Meckel's diverticulum (n = 1), respectively. The average time from symptom onset to diagnosis was 4.5 ± 4.4 years. Other vascular malformations were detected in eight patients (34.8%). All patients showed a positive anomalous vascular image on contrast-enhanced computed tomography (CT) or magnetic resonance imaging (MRI). The sensitivity of colonoscopy in the diagnosis of CRVM was 82.6% (19/23). A total of 21 patients underwent a modified Soave procedure. The lesions were mostly restricted to the colorectum and showed transmural diffuse distribution, with an average length of 20 ± 5.4 cm. Two patients (9.5%) experienced surgical complications. Bloody stools reappeared in two patients (9.5%), and colonoscopy showed abnormal angiogenesis at the anastomotic site, which were cured by sclerotherapy and/or electrocautery. The median follow-up time was 78 months. Bloody stools were absent at the last time of follow-up, and hemoglobin was in the normal range for all patients.

Conclusion The identification of CRVM in children often is delayed. Colonoscopy, CT, and MRI are essential in making the correct diagnosis. The modified Soave procedure is safe and feasible to treat CRVM in children. Endoscopic sclerotherapy and/or electrocautery can be used for residual lesions.

Keywords

vascular malformation - colorectal - children - hematochezia

* These authors contributed equally to this work and retain the first authorship.




Publikationsverlauf

Eingereicht: 05. April 2022

Angenommen: 18. Mai 2022

Artikel online veröffentlicht:
13. August 2022

© 2022. Thieme. All rights reserved.

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Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Zeng Z, Wu X, Chen J, Luo S, Hou Y, Kang L. Safety and feasibility of transanal endoscopic surgery for diffuse cavernous hemangioma of the rectum. Gastroenterol Res Pract 2019; 2019: 1732340
    CrossrefPubMedGoogle Scholar
  • 2 Wang HT, Gao XH, Fu CG, Wang L, Meng RG, Liu LJ. Diagnosis and treatment of diffuse cavernous hemangioma of the rectum: report of 17 cases. World J Surg 2010; 34 (10) 2477-2486
    CrossrefPubMedGoogle Scholar
  • 3 Vitor S, Oliveira Ferreira A, Lopes J, Velosa J. Hemangioma of the rectum - how misleading can hematochezia be?. Rev Esp Enferm Dig 2016; 108 (08) 500-501
    PubMedGoogle Scholar
  • 4 El-Sheikha J, Little MW, Bratby M. Rectal venous malformation treated by superior rectal artery embolization. Cardiovasc Intervent Radiol 2019; 42 (01) 154-157
    CrossrefPubMedGoogle Scholar
  • 5 Fishman SJ, Burrows PE, Leichtner AM, Mulliken JB. Gastrointestinal manifestations of vascular anomalies in childhood: varied etiologies require multiple therapeutic modalities. J Pediatr Surg 1998; 33 (07) 1163-1167
    CrossrefPubMedGoogle Scholar
  • 6 Lv Z, Xiao X, Zheng J, Liu J, Chen G. Modified Soave procedure for the treatment of vascular malformations involving anorectum and sigmoid colon. J Pediatr Surg 2009; 44 (12) 2359-2363
    CrossrefPubMedGoogle Scholar
  • 7 Cotzias E, Rehman SF, Arsalani Zadeh R, Smith D. Conservative management of diffuse cavernous haemangioma of the sigmoid and rectum. Ann R Coll Surg Engl 2020; 102 (01) e1-e3
    CrossrefPubMedGoogle Scholar
  • 8 Li HB, Lv JF, Lu N, Lv ZS. Mechanical intestinal obstruction due to isolated diffuse venous malformations in the gastrointestinal tract: a case report and review of literature. World J Clin Cases 2020; 8 (01) 157-167
    CrossrefPubMedGoogle Scholar
  • 9 Andrade P, Lopes S, Macedo G. Diffuse cavernous hemangioma of the rectum: case report and literature review. Int J Colorectal Dis 2015; 30 (09) 1289-1290
    CrossrefPubMedGoogle Scholar
  • 10 Komekami Y, Konishi F, Makita K. et al. Rectal arterio-venous malformation (AVM) with bleeding of an internal hemorrhoid. Clin J Gastroenterol 2016; 9 (01) 22-26
    CrossrefPubMedGoogle Scholar
  • 11 Ganesananthan S, Barlow J, Durai D, Hawthorne AB. Multiple venous malformations in the left colon and rectum: a long-standing case managed conservatively and an update of current literature. BMJ Case Rep 2019; 12 (03) e227700
    CrossrefPubMedGoogle Scholar
  • 12 de la Torre L, Carrasco D, Mora MA, Ramírez J, López S. Vascular malformations of the colon in children. J Pediatr Surg 2002; 37 (12) 1754-1757
    CrossrefPubMedGoogle Scholar
  • 13 Martinez-Lopez A, Salvador-Rodriguez L, Montero-Vilchez T, Molina-Leyva A, Tercedor-Sanchez J, Arias-Santiago S. Vascular malformations syndromes: an update. Curr Opin Pediatr 2019; 31 (06) 747-753
    CrossrefPubMedGoogle Scholar
  • 14 Masjedizadeh AR, Alavinejad P, Hashemi SJ. Rectal bleeding in a young patient with cavernous hemangioma and bone hypertrophy. Middle East J Dig Dis 2016; 8 (01) 73-74
    CrossrefPubMedGoogle Scholar
  • 15 Bhattacharjee HK, Nariampalli Karthyarth M, Suhani S. et al. Laparoscopic total mesorectal excision for rectal venous malformation: a case report with a brief literature review. Asian J Endosc Surg 2021; 14 (01) 85-89
    CrossrefPubMedGoogle Scholar
  • 16 Legiehn GM, Heran MK. Venous malformations: classification, development, diagnosis, and interventional radiologic management. Radiol Clin North Am 2008; 46 (03) 545-597 , vi
    CrossrefPubMedGoogle Scholar
  • 17 Sharma M, Adulqader A, Shifa R. Endoscopic ultrasound for cavernous hemangioma of rectum. Endosc Ultrasound 2014; 3 (01) 63-65
    CrossrefPubMedGoogle Scholar
  • 18 Markovic JN, Nag U, Shortell CK. Safety and efficacy of foam sclerotherapy for treatment of low-flow vascular malformations in children. J Vasc Surg Venous Lymphat Disord 2020; 8 (06) 1074-1082
    CrossrefPubMedGoogle Scholar
  • 19 Wang HT, Tu Y, Fu CG. et al. Diffuse cavernous hemangioma of the rectosigmoid colon. Tech Coloproctol 2005; 9 (02) 145-148
    CrossrefPubMedGoogle Scholar
  • 20 Londono-Schimmer EE, Ritchie JK, Hawley PR. Coloanal sleeve anastomosis in the treatment of diffuse cavernous haemangioma of the rectum: long-term results. Br J Surg 1994; 81 (08) 1235-1237
    CrossrefPubMedGoogle Scholar
  • 21 Wu XR, Liang WW, Zhang XW, Kang L, Lan P. Transanal total mesorectal excision as a surgical procedure for diffuse cavernous hemangioma of the rectum: a case report. Int J Surg Case Rep 2017; 39: 164-167
    CrossrefPubMedGoogle Scholar
  • 22 Anupama B, Zheng S, Xiao X. Ten-year experience in the management of total colonic aganglionosis. J Pediatr Surg 2007; 42 (10) 1671-1676
    CrossrefPubMedGoogle Scholar
  • 23 Sierra A, Baixauli J, Elosúa T, Arredondo J, Cienfuegos JA. Life-threatening hematochezia from a rectal venous malformation successfully treated with microfoam sclerotherapy: description of a new technique. Int J Colorectal Dis 2015; 30 (10) 1427-1428
    CrossrefPubMedGoogle Scholar
  • 24 Gurien LA, Jackson RJ, Kiser MM, Richter GT. Nd:YAG laser therapy for rectal and vaginal venous malformations. Pediatr Surg Int 2017; 33 (08) 887-891
    CrossrefPubMedGoogle Scholar
  • 25 Lee TY, Yeh HZ, Yang SS, Chang CS. Successful application of haemoclips plus argon plasma coagulation for angioectasia bleeding with underlying arteriovenous malformation in the colon. Colorectal Dis 2010; 12 (7 Online): e180-e181
    PubMedGoogle Scholar
  • 26 Abematsu T, Okamoto Y, Nakagawa S. et al. Rectosigmoid colon venous malformation successfully treated with propranolol and celecoxib. J Pediatr Surg Case Rep 2015; 3 (08) 331-333
    CrossrefPubMedGoogle Scholar