International Journal of Hematology

, Volume 97, Issue 4, pp 529–534 | Cite as

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

  • Masahiro Hirayama
  • Eiichi AzumaEmail author
  • Atsuko Nakazawa
  • Shotaro Iwamoto
  • Hidemi Toyoda
  • Yoshihiro Komada
Case Report


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.


Gastric antral vascular ectasia Protein-losing enteropathy Hematopoietic stem cell transplantation Graft-versus-host disease 



Written informed consent to publication was obtained.

Conflict of interest

There are no conflicts of interest in the all authors.


  1. 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.PubMedCrossRefGoogle Scholar
  2. 2.
    Selinger CP, Ang YS. Gastric antral vascular ectasia (GAVE): an update on clinical presentation, pathophysiology and treatment. Digestion. 2008;77:131–7.PubMedCrossRefGoogle Scholar
  3. 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.PubMedGoogle Scholar
  4. 4.
    Barnett JL. Images in clinical medicine. Vascular ectasia of the gastric antrum. New Engl J Med. 1998;339:166.PubMedCrossRefGoogle Scholar
  5. 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.PubMedGoogle Scholar
  6. 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.PubMedCrossRefGoogle Scholar
  7. 7.
    Al-Mogairen SM. Lupus protein-losing enteropathy (LUPLE): a systematic review. Rheumatol Int. 2011;31:995–1001.PubMedCrossRefGoogle Scholar
  8. 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.PubMedCrossRefGoogle Scholar
  9. 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.PubMedCrossRefGoogle Scholar
  10. 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.PubMedCrossRefGoogle Scholar
  11. 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.PubMedCrossRefGoogle Scholar
  12. 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.PubMedCrossRefGoogle Scholar
  13. 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.PubMedCrossRefGoogle Scholar
  14. 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.PubMedCrossRefGoogle Scholar
  15. 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.PubMedCrossRefGoogle Scholar
  16. 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.PubMedCrossRefGoogle Scholar
  17. 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.PubMedCrossRefGoogle Scholar
  18. 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.PubMedGoogle Scholar
  19. 19.
    Calam J, Walker RJ. Antral vascular lesion, achlorhydria, and chronic gastrointestinal blood loss: response to steroids. Dig Dis Sci. 1980;25:236–9.PubMedCrossRefGoogle Scholar
  20. 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.PubMedCrossRefGoogle Scholar
  21. 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.PubMedCrossRefGoogle Scholar
  22. 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.PubMedCrossRefGoogle Scholar
  23. 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.PubMedCrossRefGoogle Scholar
  24. 24.
    Hosking SW, Kennedy HJ, Seddon I, Triger DR. The role of propranolol in congestive gastropathy of portal hypertension. Hepatology. 1987;7:437–41.PubMedCrossRefGoogle Scholar
  25. 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.PubMedCrossRefGoogle Scholar
  26. 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.PubMedGoogle Scholar
  27. 27.
    McCormick PA, Ooi H, Crosbie O. Tranexamic acid for severe bleeding gastric antral vascular ectasia in cirrhosis. Gut. 1998;42:750–2.PubMedCrossRefGoogle Scholar
  28. 28.
    Aractingi S, Chosidow O. Cutaneous graft-versus-host disease. Arch Dermatol. 1998;134:602–12.PubMedCrossRefGoogle Scholar
  29. 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.PubMedCrossRefGoogle Scholar
  30. 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.PubMedCrossRefGoogle Scholar
  31. 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.PubMedCrossRefGoogle Scholar
  32. 32.
    Umar SB, DiBaise JK. Protein-losing enteropathy: case illustrations and clinical review. Am J Gastroenterol. 2010;105:43–9. quiz 50.PubMedCrossRefGoogle Scholar
  33. 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.PubMedCrossRefGoogle Scholar
  34. 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.PubMedGoogle Scholar
  35. 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.PubMedCrossRefGoogle Scholar
  36. 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.PubMedCrossRefGoogle Scholar
  37. 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.PubMedCrossRefGoogle Scholar
  38. 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.PubMedCrossRefGoogle Scholar

Copyright information

© The Japanese Society of Hematology 2013

Authors and Affiliations

  • Masahiro Hirayama
    • 1
  • Eiichi Azuma
    • 2
    Email author
  • Atsuko Nakazawa
    • 3
  • Shotaro Iwamoto
    • 1
  • Hidemi Toyoda
    • 1
  • Yoshihiro Komada
    • 1
  1. 1.Department of PediatricsMie University Graduate School of MedicineTsuJapan
  2. 2.Department of Cell TransplantationMie University Graduate School of MedicineTsuJapan
  3. 3.Department of PathologyNational Center for Child Health and DevelopmentSetagayaJapan

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