Skip to main content

Advertisement

Log in

Simultaneous occurrence of gastric antral vascular ectasia and protein-losing enteropathy in chronic graft-versus-host disease

  • Case Report
  • Published:
International Journal of Hematology Aims and scope Submit manuscript

Abstract

Gastric antral vascular ectasia (GAVE) leading to upper gastrointestinal bleeding is a heterogeneous disorder that is not commonly recognized in hematopoietic stem cell transplantation (HSCT). Protein-losing enteropathy (PLE) is noted as another gastrointestinal complication in the context of chronic graft-versus-host disease (GVHD) after HSCT. The possibility of a relationship between these two distinct gastrointestinal disorders, however, remains obscure. A 6-year-old boy with acute myelogenous leukemia developed severe hematemesis 4 months after myeloablative HSCT from a human leukocyte antigen-matched sibling donor. The diagnosis of GAVE was made by upper endoscopy and histological examination. The patient simultaneously developed frequent diarrhea and significant hypoproteinemia, consistent with a diagnosis of PLE. This co-occurrence of GAVE and PLE against a background of chronic GVHD was successfully treated with cyclosporin A and prednisolone. To our knowledge, this is the first report of GAVE concurrent with PLE following HSCT. The possible association of GAVE and PLE in chronic GVHD is discussed.

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

Access this article

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

Instant access to the full article PDF.

Fig. 1
Fig. 2

References

  1. Papadopoulou A, Lloyd DR, Williams MD, Darbyshire PJ, Booth IW. Gastrointestinal and nutritional sequelae of bone marrow transplantation. Arch Dis Child. 1996;75:208–13.

    Article  PubMed  CAS  Google Scholar 

  2. Selinger CP, Ang YS. Gastric antral vascular ectasia (GAVE): an update on clinical presentation, pathophysiology and treatment. Digestion. 2008;77:131–7.

    Article  PubMed  Google Scholar 

  3. Lanzafame S, Torrisi A, Nesi L, Russo V, Cimino G, Rumeo A, et al. Gastric antral vascular ectasia (the watermelon stomach): a brief case report. Hepatogastroenterology. 1998;45:606–8.

    PubMed  CAS  Google Scholar 

  4. Barnett JL. Images in clinical medicine. Vascular ectasia of the gastric antrum. New Engl J Med. 1998;339:166.

    Article  PubMed  CAS  Google Scholar 

  5. Marmaduke DP, Greenson JK, Cunningham I, Herderick EE, Cornhill JF. Gastric vascular ectasia in patients undergoing bone marrow transplantation. Am J Clin Pathol. 1994;102:194–8.

    PubMed  CAS  Google Scholar 

  6. Tobin RW, Hackman RC, Kimmey MB, Durtschi MB, Hayashi A, Malik R, et al. Bleeding from gastric antral vascular ectasia in marrow transplant patients. Gastrointest Endosc. 1996;44:223–9.

    Article  PubMed  CAS  Google Scholar 

  7. Al-Mogairen SM. Lupus protein-losing enteropathy (LUPLE): a systematic review. Rheumatol Int. 2011;31:995–1001.

    Article  PubMed  CAS  Google Scholar 

  8. Ward EM, Raimondo M, Rosser BG, Wallace MB, Dickson RD. Prevalence and natural history of gastric antral vascular ectasia in patients undergoing orthotopic liver transplantation. J Clin Gastroenterol. 2004;38:898–900.

    Article  PubMed  Google Scholar 

  9. Gostout CJ, Viggiano TR, Ahlquist DA, Wang KK, Larson MV, Balm R. The clinical and endoscopic spectrum of the watermelon stomach. J Clin Gastroenterol. 1992;15:256–63.

    Article  PubMed  CAS  Google Scholar 

  10. Sebastian S, O’Morain CA, Buckley MJ. Review article: current therapeutic options for gastric antral vascular ectasia. Aliment Pharmacol Ther. 2003;18:157–65.

    Article  PubMed  CAS  Google Scholar 

  11. Selinger RR, McDonald GB, Hockenbery DM, Steinbach G, Kimmey MB. Efficacy of neodymium:YAG laser therapy for gastric antral vascular ectasia (GAVE) following hematopoietic cell transplant. Bone Marrow Transplant. 2006;37:191–7.

    Article  PubMed  CAS  Google Scholar 

  12. Ohashi K, Sanaka M, Tu Y, Egawa N, Funata N, Okuyama Y, et al. Clinical features and treatment of hematopoietic stem cell transplantation-associated gastric antral vascular ectasia. Bone Marrow Transplant. 2003;32:417–21.

    Article  PubMed  CAS  Google Scholar 

  13. Kuroiwa Y, Suzuki N, Mizue N, Hori T, Endo T, Yoshida Y, et al. Gastric antral vascular ectasia in 2-yr-old girl undergoing unrelated cord blood stem cell transplantation. Pediatr Transplant. 2005;9:788–91.

    Article  PubMed  Google Scholar 

  14. Congly SE, Gao ZH, Storr M. Duodenal vascular ectasia (DUVE) associated with hematopoietic stem cell transplant. Endoscopy. 2011;43(Suppl 2 UCTN):E197–8.

    Article  PubMed  Google Scholar 

  15. Ikeda M, Hayashi N, Imamura E, Kaneko A, Michida T, Yamamoto K, et al. Endoscopic follow-up study of development of gastric antral vascular ectasia associated with liver cirrhosis. J Gastroenterol. 1997;32:587–92.

    Article  PubMed  CAS  Google Scholar 

  16. Yamada M, Nishimura D, Hoshino H, Katada N, Sano H, Kato K. Gastric antral vascular ectasia successfully treated by endoscopic electrocoagulation. J Gastroenterol. 1998;33:546–9.

    Article  PubMed  CAS  Google Scholar 

  17. Spahr L, Villeneuve JP, Dufresne MP, Tasse D, Bui B, Willems B, et al. Gastric antral vascular ectasia in cirrhotic patients: absence of relation with portal hypertension. Gut. 1999;44:739–42.

    Article  PubMed  CAS  Google Scholar 

  18. Jabbari M, Cherry R, Lough JO, Daly DS, Kinnear DG, Goresky CA. Gastric antral vascular ectasia: the watermelon stomach. Gastroenterology. 1984;87:1165–70.

    PubMed  CAS  Google Scholar 

  19. Calam J, Walker RJ. Antral vascular lesion, achlorhydria, and chronic gastrointestinal blood loss: response to steroids. Dig Dis Sci. 1980;25:236–9.

    Article  PubMed  CAS  Google Scholar 

  20. Lorenzi AR, Johnson AH, Davies G, Gough A. Gastric antral vascular ectasia in systemic sclerosis: complete resolution with methylprednisolone and cyclophosphamide. Ann Rheum Dis. 2001;60:796–8.

    Article  PubMed  CAS  Google Scholar 

  21. Moss SF, Ghosh P, Thomas DM, Jackson JE, Calam J. Gastric antral vascular ectasia: maintenance treatment with oestrogen-progesterone. Gut. 1992;33:715–7.

    Article  PubMed  CAS  Google Scholar 

  22. Tran A, Villeneuve JP, Bilodeau M, Willems B, Marleau D, Fenyves D, et al. Treatment of chronic bleeding from gastric antral vascular ectasia (GAVE) with estrogen-progesterone in cirrhotic patients: an open pilot study. Am J Gastroenterol. 1999;94:2909–11.

    Article  PubMed  CAS  Google Scholar 

  23. Dunne KA, Hill J, Dillon JF. Treatment of chronic transfusion-dependent gastric antral vascular ectasia (watermelon stomach) with thalidomide. Eur J Gastroenterol Hepatol. 2006;18:455–6.

    Article  PubMed  Google Scholar 

  24. Hosking SW, Kennedy HJ, Seddon I, Triger DR. The role of propranolol in congestive gastropathy of portal hypertension. Hepatology. 1987;7:437–41.

    Article  PubMed  CAS  Google Scholar 

  25. Perez-Ayuso RM, Pique JM, Bosch J, Panes J, Gonzalez A, Perez R, et al. Propranolol in prevention of recurrent bleeding from severe portal hypertensive gastropathy in cirrhosis. Lancet. 1991;337:1431–4.

    Article  PubMed  CAS  Google Scholar 

  26. Disdier P, Schleinitz N, Perreard M, Monges D, Swiader L, Gerolami A, et al. Dramatic improvement of watermelon stomach with alpha-interferon. Am J Gastroenterol. 1995;90:1009–10.

    PubMed  CAS  Google Scholar 

  27. McCormick PA, Ooi H, Crosbie O. Tranexamic acid for severe bleeding gastric antral vascular ectasia in cirrhosis. Gut. 1998;42:750–2.

    Article  PubMed  CAS  Google Scholar 

  28. Aractingi S, Chosidow O. Cutaneous graft-versus-host disease. Arch Dermatol. 1998;134:602–12.

    Article  PubMed  CAS  Google Scholar 

  29. Soares AB, Faria PR, Magna LA, Correa ME, de Sousa CA, Almeida OP, et al. Chronic GVHD in minor salivary glands and oral mucosa: histopathological and immunohistochemical evaluation of 25 patients. J Oral Pathol Med. 2005;34:368–73.

    Article  PubMed  CAS  Google Scholar 

  30. Favre A, Cerri A, Bacigalupo A, Lanino E, Berti E, Grossi CE. Immunohistochemical study of skin lesions in acute and chronic graft versus host disease following bone marrow transplantation. Am J Surg Pathol. 1997;21:23–34.

    Article  PubMed  CAS  Google Scholar 

  31. Hirayama M, Azuma E, Nakagawa-Nakazawa A, Kumamoto T, Iwamoto S, Amano K, et al. Interleukin-10 spot-forming cells as a novel biomarker of chronic graft-versus-host disease. Haematologica. 2013;98:41–9.

    Article  PubMed  CAS  Google Scholar 

  32. Umar SB, DiBaise JK. Protein-losing enteropathy: case illustrations and clinical review. Am J Gastroenterol. 2010;105:43–9. quiz 50.

    Article  PubMed  Google Scholar 

  33. Awazawa R, Yamamoto Y, Mine Y, Nakamura I, Kishimoto K, Kinjyo F, et al. Systemic lupus erythematosus complicated with protein-losing enteropathy: a case report and review of the published works. J Dermatol. 2012;39:454–61.

    Article  PubMed  CAS  Google Scholar 

  34. Pinho Vaz C, Ibrahim A, Avila Garavito A, Vantelon JM, Bourhis JH, Charnoze I, et al. Protein-losing gastropathy associated with cytomegalovirus: a rare and late complication of allogeneic bone marrow transplantation. Bone Marrow Transplant. 1996;17:887–9.

    PubMed  CAS  Google Scholar 

  35. Mok CC, Ying KY, Mak A, To CH, Szeto ML. Outcome of protein-losing gastroenteropathy in systemic lupus erythematosus treated with prednisolone and azathioprine. Rheumatology. 2006;45:425–9.

    Article  PubMed  CAS  Google Scholar 

  36. Kim YG, Lee CK, Byeon JS, Myung SJ, Oh JS, Nah SS, et al. Serum cholesterol in idiopathic and lupus-related protein-losing enteropathy. Lupus. 2008;17:575–9.

    Article  PubMed  CAS  Google Scholar 

  37. Akpek G, Chinratanalab W, Lee LA, Torbenson M, Hallick JP, Anders V, et al. Gastrointestinal involvement in chronic graft-versus-host disease: a clinicopathologic study. Biol Blood Marrow Transplant. 2003;9:46–51.

    Article  PubMed  Google Scholar 

  38. Tuncer HH, Rana N, Milani C, Darko A, Al-Homsi SA. Gastrointestinal and hepatic complications of hematopoietic stem cell transplantation. World J Gastroenterol. 2012;18:1851–60.

    Article  PubMed  Google Scholar 

Download references

Acknowledgments

Written informed consent to publication was obtained.

Conflict of interest

There are no conflicts of interest in the all authors.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Eiichi Azuma.

About this article

Cite this article

Hirayama, M., Azuma, E., Nakazawa, A. et al. Simultaneous occurrence of gastric antral vascular ectasia and protein-losing enteropathy in chronic graft-versus-host disease. Int J Hematol 97, 529–534 (2013). https://doi.org/10.1007/s12185-013-1279-y

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12185-013-1279-y

Keywords

Navigation