Skip to main content
SpringerLink
Log in

Clipping with double-balloon endoscopy for small intestinal venous malformations in a patient with blue rubber bleb nevus syndrome

  • Case Report
  • Published:
Clinical Journal of Gastroenterology Aims and scope Submit manuscript

Abstract

Blue rubber bleb nevus syndrome (BRBNS) is a rare syndrome characterized by venous malformations in the skin and gastrointestinal tract, especially in the small intestine. Patients with BRBNS have increased risks of gastrointestinal hemorrhage and anemia. This is the first report in the English literature on BRBNS with hemangiomas in the small intestine that were treated successfully by endoscopic clipping using double-balloon endoscopy. A 25-year-old Japanese man presented to a local clinic with dyspnea, fatigue, and a hemoglobin level < 5 g/dL. The diagnosis was iron deficiency anemia. Since childhood, he had had a hemangioma in the shoulder joint and hemangiomas in the skin on the left arm. However, neither upper nor lower gastrointestinal endoscopy showed any lesions and he was referred to us for further evaluation and treatment of the anemia. Small bowel capsule endoscopy (SBCE) revealed hemangiomas in the small intestine, one of which was bleeding. Transanal DBE revealed a 10-mm bluish-purple hemangioma with erosion on the surface, which became smaller after application of five clips. Follow-up SBCE on day 50 showed that the hemangioma had completely disappeared. Clipping may be a safe and effective treatment for small bowel hemangioma in BRBNS.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

Abbreviations

BRBNS:

Blue rubber bleb nevus syndrome

DBE:

Double-balloon endoscopy

SBCE:

Small bowel capsule endoscopy

References

  1. Isoldi S, Belsha D, Yeop I, et al. Diagnosis and management of children with blue rubber bleb nevus syndrome: a multi-center case series. Dig Liver Dis. 2019;51:1537–46.

    Article  Google Scholar 

  2. Jin XL, Wang ZH, Xiao XB, et al. Blue rubber bleb nevus syndrome: a case report and literature review. World J Gastroenterol. 2014;20:17254–9.

    Article  Google Scholar 

  3. Barlas A, Avsar E, Bozbas A, et al. Role of capsule endoscopy in blue rubber bleb nevus syndrome. Can J Surg. 2008;51:E119–20.

    PubMed  PubMed Central  Google Scholar 

  4. Bean WB. Vascular spiders and related lesions of the skin. Springfield: Charles C Thomas Publisher; 1958. p. 178–85.

    Google Scholar 

  5. Gallo SH, McClave SA. Blue rubber bleb nevus syndrome: gastrointestinal involvement and its endoscopic presentation. Gastrointest Endosc. 1992;38:72–6.

    Article  CAS  Google Scholar 

  6. Gallione CJ, Pasyk KA, Boon LM, et al. A gene for familial venous malformations maps to chromosome 9p in a second large kindred. J Med Genet. 1995;32:197–9.

    Article  CAS  Google Scholar 

  7. Rodrigues D, Bourroul ML, Ferrer AP, et al. Blue rubber bleb nevus syndrome. Rev Hosp Clin Fac Med Sao Paulo. 2000;55:29–34.

    Article  CAS  Google Scholar 

  8. Mikami S, Matsumoto Y, Yamada S, et al. The first case of Blue rubber bleb nevus syndrome whose intestinal lesions were successfully treated by clipping therapy using single balloon enteroscopy. Gastroenterol Endosc. 2011;53:275–82.

    Google Scholar 

  9. Mensink PBF, Haringsma J, Kucharzik T, et al. Complications of double balloon enteroscopy: a multicenter survey. Endoscopy. 2007;39:613–5.

    Article  CAS  Google Scholar 

  10. Gao Z, Zhang Y, Li W, et al. Effectiveness and safety of polidocanol for the treatment of hemangiomas and vascular malformations: a meta-analysis. Dermatol Ther. 2018. https://doi.org/10.1111/dth.12568.

    Article  PubMed  Google Scholar 

  11. Malafaia MC, Heissat S, Jacob P, et al. Blue rubber bleb nevus syndrome: endoscopic treatment with sclerotherapy during double-balloon enteroscopy in a 9-year-old boy. Endoscopy. 2019;51:E98–100.

    Article  Google Scholar 

  12. Marakhouski K, Sharafanovich E, Kolbik U, et al. Endoscopic treatment of blue rubber bleb nevus syndrome in a 4-year-old girl with long-term follow-up: a case report. World J Gastrointest Endosc. 2021;13:90–6.

    Article  Google Scholar 

  13. Aoyama T, Fukumoto A, Shigita K, et al. Successful endoscopic sclerotherapy using polidocanol for small bowel hemangioma. Intern Med. 2020;59:1727–30.

    Article  Google Scholar 

  14. Wang Z, Yang X, Wu L, et al. Blue rubber bleb nevus syndrome: treatment of lesions in the small intestine with repeated injection of lauromacrogol. Gastrointest Endosc. 2015;81:1274–5.

    Article  Google Scholar 

  15. Igawa A, Oka S, Tanaka S, et al. Polidocanol injection therapy for small-bowel hemangioma by using double-balloon endoscopy. Gastrointest Endosc. 2016;84:163–7.

    Article  Google Scholar 

  16. Grupka MJ, Benson J. Endoscopic clipping. J Dig Dis. 2008;9:72–8.

    Article  Google Scholar 

  17. De Bona M, Bellumat A, De Boni M. Capsule endoscopy for the diagnosis and follow-up of blue rubber bleb nevus syndrome. Dig Liver Dis. 2005;37:451–3.

    Article  Google Scholar 

  18. Thomson M, Venkatesh K, Elmalik K, et al. Double balloon enteroscopy in children: diagnosis, treatment, and safety. World J Gastroenterol. 2010;16:56–62.

    PubMed  PubMed Central  Google Scholar 

  19. Jin J, Pan J, Zhu L. Therapy for hemangiomas of blue rubber bleb nevus syndrome in the small intestine with single balloon endoscopy. Dig Endosc. 2015;27:780.

    Article  Google Scholar 

  20. Rubio-Mateos JM, Tojo-González R, Pérez-Cuadrado-Robles E. Endoscopic mucosal resection by double-balloon enteroscopy can be an alternative in small bowel venous malformations. Dig Endosc. 2018;30:789.

    Article  Google Scholar 

  21. Lazaridis N, Murino A, Koukias N, et al. Blue rubber bleb nevus syndrome in a 10-year-old child treated with loop ligation facilitated by double-balloon enteroscopy. VideoGIE. 2020;5:412–4.

    Article  Google Scholar 

  22. Dooghaie Moghadam A, Bagheri M, Eslami P, et al. Blue rubber bleb nevus syndrome because of 12 years of iron deficiency anemia in a patient by double balloon enteroscopy; A case report and review of literature. Middle East J Dig Dis. 2021;13:153–9.

    Article  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Gastroenterology, Toranomon Hospital, 2-2-2 Toranomon, Minato-ku, Tokyo, 105-8470, Japan

    Kumi Nitta, Akira Matsui, Daisuke Kikuchi & Shu Hoteya

  2. Toranomon Hospital Health Management Center, 1-8-1 Akasaka, Minato-ku, Tokyo, 107-0052, Japan

    Akihiro Araki

Authors
  1. Kumi Nitta
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Akira Matsui
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Akihiro Araki
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Daisuke Kikuchi
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Shu Hoteya
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Akira Matsui.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Human/animal rights

All procedure followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2013.

Informed consent

Informed consent was obtained from all patients included in the study.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Nitta, K., Matsui, A., Araki, A. et al. Clipping with double-balloon endoscopy for small intestinal venous malformations in a patient with blue rubber bleb nevus syndrome. Clin J Gastroenterol 15, 901–906 (2022). https://doi.org/10.1007/s12328-022-01670-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12328-022-01670-0

Keywords

Search

Navigation