Unrelated bone marrow transplantation improves Crohn's disease in a patient with myelodysplastic syndrome that developed from aplastic anemia: A case report

Goto,Hideko; Hara,Takeshi; Fujita,Kei; Kaneda,Yuto; Yamaguchi,Kimihiro; Shibata,Yuhei; Kasahara,Senji; Takahashi,Takeshi; Tsurumi,Hisashi

doi:10.3892/wasj.2022.147

Unrelated bone marrow transplantation improves Crohn's disease in a patient with myelodysplastic syndrome that developed from aplastic anemia: A case report

Authors:
- Hideko Goto
- Takeshi Hara
- Kei Fujita
- Yuto Kaneda
- Kimihiro Yamaguchi
- Yuhei Shibata
- Senji Kasahara
- Takeshi Takahashi
- Hisashi Tsurumi
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Affiliations: Department of Hematology, Gifu Municipal Hospital, Gifu 500‑8513, Japan, Department of Hematology, Matsunami General Hospital, Gifu 501‑6062, Japan
Published online on: March 1, 2022 https://doi.org/10.3892/wasj.2022.147
Article Number: 12
Copyright: © Goto et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

The present study describes the case of a 56‑year‑old male who was admitted to hospital due to pancytopenia. He was diagnosed as having acquired severe aplastic anemia (SAA). Immunosuppressive therapy with cyclosporine was transiently effective; however, the pancytopenia recurred. Bone marrow aspiration did not reveal an increased number of blasts, although it revealed trilineage dysplasia. Chromosomal analysis of the bone marrow demonstrated der(1;7) with abnormalities on chromosome 1. A diagnosis of a transformation from SAA to myelodysplastic syndrome (MDS) was made. High‑grade fever and lower abdominal pain developed at approximately the same time. The patient was diagnosed as having Crohn's disease (CD). Treatment consisting of mesalazine and adalimumab was commenced. After two courses of azacitidine treatment, allogeneic bone marrow transplantation was performed, preceded by a reduced‑intensity conditioning regimen, including fludarabine, cytarabine and cyclophosphamide. Tacrolimus and short‑term methotrexate were used as prophylaxis against graft‑versus‑host disease. Chromosomal analysis of the bone marrow revealed a 100% donor‑derived abnormal karyotype. The patient has since then remained in complete remission of both MDS and CD. On the whole, the present study demonstrates that allogeneic hematopoietic stem cell transplantation may be a promising treatment strategy for patients with combined MDS and CD. However, its use should be carefully considered.

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1	Young NS, Calado RT and Scheinberg P: Current concepts in the pathophysiology and treatment of aplastic anemia. Blood. 108:2509–2519. 2006.PubMed/NCBI View Article : Google Scholar
2	Tefferi A and Vardiman JW: Myelodysplastic syndromes. N Engl J Med. 361:1872–1885. 2009.PubMed/NCBI View Article : Google Scholar
3	Wang Z, Zhou Y and Liu Y: Concurrent inflammatory bowel disease and myelodysplastic syndrome: Report of nine new cases and a review of the literature. Dig Dis Sci. 53:1929–1932. 2008.PubMed/NCBI View Article : Google Scholar
4	Eng C, Farraye FA, Shulman LN, Peppercorn MA, Krauss CM, Connors JM and Stone RM: The association between the myelodysplastic syndromes and Crohn disease. Ann Intern Med. 117:661–662. 1992.PubMed/NCBI View Article : Google Scholar
5	Castellote J, Porta F, Tuset E and Salinas R: Crohn's disease and the myelodysplastic syndrome. J Clin Gastroenterol. 24:286–287. 1997.PubMed/NCBI View Article : Google Scholar
6	Bosch X, Bernadich O and Vera M: The association between Crohn disease and the myelodysplastic syndromes. Report of 3 cases and review of the literature. Medicine (Baltimore). 77:371–377. 1998.PubMed/NCBI View Article : Google Scholar
7	Wang H, Chuhjo T, Yamazaki H, Shiobara S, Teramura M, Mizoguchi H and Nakao S: Relative increase of granulocytes with a paroxysmal nocturnal haemoglobinuria phenotype in aplastic anaemia patients: The high prevalence at diagnosis. Eur J Haematol. 66:200–205. 2001.PubMed/NCBI View Article : Google Scholar
8	Greenberg PL, Tuechler H, Schanz J, Sanz G, Garcia-Manero G, Solé F, Bennett JM, Bowen D, Fenaux P, Dreyfus F, et al: Revised international prognostic scoring system for myelodysplastic syndromes. Blood. 120:2454–2465. 2012.PubMed/NCBI View Article : Google Scholar
9	Malcovati L, Germing U, Kuendgen A, Della Porta MG, Pascutto C, Invernizzi R, Giagounidis A, Hildebrandt B, Bernasconi P, Knipp S, et al: Time-dependent prognostic scoring system for predicting survival and leukemic evolution in myelodysplastic syndromes. J Clin Oncol. 25:3503–3510. 2007.PubMed/NCBI View Article : Google Scholar
10	Hiraoka A, Ohashi Y, Okamoto S, Moriyama Y, Nagao T, Kodera Y, Kanamaru A, Dohy H and Masaoka T: Japanese FK506 BMT(Bone Marrow Transplantation) Study Group. Phase III study comparing tacrolimus (FK506) with cyclosporine for graft-versus-host disease prophylaxis after allogeneic bone marrow transplantation. Bone Marrow Transplant. 28:181–185. 2001.PubMed/NCBI View Article : Google Scholar
11	Sun L and Babushok DV: Secondary myelodysplastic syndrome and leukemia in acquired aplastic anemia and paroxysmal nocturnal hemoglobinuria. Blood. 136:36–49. 2020.PubMed/NCBI View Article : Google Scholar
12	Saif MW, Hopkins JL and Gore SD: Autoimmune phenomena in patients with myelodysplastic syndromes and chronic myelomonocytic leukemia. Leuk Lymphoma. 43:2083–2092. 2002.PubMed/NCBI View Article : Google Scholar
13	Braun T and Fenaux P: Myelodysplastic syndromes (MDS) and autoimmune disorders (AD): cause or consequence? Best Pract Res Clin Haematol. 26:327–336. 2013.PubMed/NCBI View Article : Google Scholar
14	Shimamoto T and Ohyashiki K: Immunosuppressive treatments for myelodysplastic syndromes. Leuk Lymphoma. 44:593–604. 2003.PubMed/NCBI View Article : Google Scholar
15	Hanauer SB, Wong KK, Frank PH, Sweet DL and Kirsner JB: Acute leukemia following inflammatory bowel disease. Dig Dis Sci. 27:545–548. 1982.PubMed/NCBI View Article : Google Scholar
16	Nahas SC, Nahas CS, Marques CF, Borba MR, Helito AS and Odoni V: Concurrent development of Crohn disease and myelodysplastic syndrome in a child: Case report and literature review. Pediatr Hematol Oncol. 23:477–483. 2006.PubMed/NCBI View Article : Google Scholar
17	Harewood GC, Loftus EV Jr, Tefferi A, Tremaine WJ and Sandborn WJ: Concurrent inflammatory bowel disease and myelodysplastic syndromes. Inflamm Bowel Dis. 5:98–103. 1999.PubMed/NCBI View Article : Google Scholar
18	Hebbar M, Kozlowski D, Wattel E, Mastrini S, Diévart M, Duclos B, Bonaz B, d'Almagne H, Belaiche J, Colombel JF and Fenaux P: Association between myelodysplastic syndromes and inflammatory bowel diseases. Report of seven new cases and review of the literature. Leukemia. 11:2188–2191. 1997.PubMed/NCBI View Article : Google Scholar
19	Hamblin T: Immunologic abnormalities in myelodysplastic syndromes. Hematol Oncol Clin North Am. 6:571–586. 1992.PubMed/NCBI
20	Molldrem JJ, Jiang YZ, Stetler-Stevenson M, Mavroudis D, Hensel N and Barrett AJ: Haematological response of patients with myelodysplastic syndrome to antithymocyte globulin is associated with a loss of lymphocyte-mediated inhibition of CFU-GM and alterations in T-cell receptor Vbeta profiles. Br J Haematol. 102:1314–1322. 1998.PubMed/NCBI View Article : Google Scholar
21	Hu C, Lv L, Liu D and Huo J: Treatment of Crohn's disease complicated with myelodysplastic syndrome via allogeneic hematopoietic stem cell transplantation: Case report and literature review. Clin J Gastroenterol. 7:299–304. 2014.PubMed/NCBI View Article : Google Scholar
22	Passweg JR, Baldomero H, Bregni M, Cesaro S, Dreger P, Duarte RF, Falkenburg JH, Kröger N, Farge-Bancel D, Gaspar HB, et al: Hematopoietic SCT in Europe: Data and trends in 2011. Bone Marrow Transplant. 48:1161–1167. 2013.PubMed/NCBI View Article : Google Scholar
23	Duijvestein M, van den Brink GR and Hommes DW: Stem cells as potential novel therapeutic strategy for inflammatory bowel disease. J Crohns Colitis. 2:99–106. 2008.PubMed/NCBI View Article : Google Scholar
24	Ditschkowski M, Einsele H, Schwerdtfeger R, Bunjes D, Trenschel R, Beelen DW and Elmaagacli AH: Improvement of inflammatory bowel disease after allogeneic stem-cell transplantation. Transplantation. 75:1745–1747. 2003.PubMed/NCBI View Article : Google Scholar
25	Sorror ML, Maris MB, Storb R, Baron F, Sandmaier BM, Maloney DG and Storer B: Hematopoietic cell transplantation (HCT)-specific comorbidity index: A new tool for risk assessment before allogeneic HCT. Blood. 106:2912–2919. 2005.PubMed/NCBI View Article : Google Scholar