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Vitiligo following stem-cell transplant

Bone Marrow Transplantation volume 55, pages 332–340 (2020)Cite this article

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Abstract

Vitiligo is an acquired skin disorder characterised by depigmentation of skin due to the selective destruction of melanocytes. The pathogenesis of vitiligo is still relatively unknown, but associations between vitiligo and other systemic autoimmune diseases have been made, such as with patients who have undergone haematopoietic stem cell transplantation (HSCT) in the treatment of haematological conditions. This article aims to review published literature that have reported the incidence of vitiligo following HSCT in order to understand their relationship, and any potential connections between vitiligo and graft-versus-host disease (GVHD). On the 21st of May 2017, the databases Pubmed, EMBASE and SCOPUS were searched using the search terms Stem cell or transplant or GVHD AND vitiligo or leucoderma or leucoderma. There was a total of 38 cases (from 28 case report articles) along with 9 cohort studies that were included in the review. The studies reviewed showed that the incidence of vitiligo in patients who have received HSCT is higher than that of the normal population. Current studies are largely limited to single-case reports, which have certain inconsistencies with regard to patient/donor profiling, and future studies should look to address these issues.

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References

  1. Alikhan A, Felsten LM, Daly M, Petronic-Rosic V. Vitiligo: a comprehensive overview Part I. Introduction, epidemiology, quality of life, diagnosis, differential diagnosis, associations, histopathology, etiology, and work-up. J Am Acad Dermatol. 2011;65:473–91.

    PubMed  Google Scholar 

  2. Byrne M, Savani BN, Mohty M, Nagler A. Peripheral blood stem cell versus bone marrow transplantation: A perspective from the Acute Leukemia Working Party of the European Society for Blood and Marrow Transplantation. Exp Hematol. 2016;44:567–73.

    PubMed  Google Scholar 

  3. Bae JM, Choi KH, Jung HM, Kim SY, Kim M, Kim GM, et al. Subsequent vitiligo after hematopoietic stem cell transplantation: a nationwide population-based cohort study from Korea. J Am Acad Dermatol. 2017;76:459–63.

    PubMed  Google Scholar 

  4. Alajlan A, Alfadley A, Pedersen K-T. Transfer of vitiligo after allogeneic bone marrow transplantation. J Am Acad Dermatol. 2002;46:606–10.

    PubMed  Google Scholar 

  5. Au WY, Yeung CK, Chan HH, Lie AK. Generalized vitiligo after lymphocyte infusion for relapsed leukaemia. Br J Dermatol. 2001;145:1015–7.

    CAS  PubMed  Google Scholar 

  6. Campbell-Fontaine A, Coad JE, Kovach R, Ericson SG. Adoptive transfer of vitiligo after allogeneic peripheral blood stem cell transplant. Bone Marrow Transpl. 2005;36:745–6.

    CAS  Google Scholar 

  7. Cathcart S, Morrell D. Vitiligo as a post-bone marrow transplantation complication. J Pediatr Hematol/Oncol. 2007;29:485–7.

    Google Scholar 

  8. Cho SB, Roh MR, Chung KY, Lee K-H, Park Y-K. Generalized vitiligo after allogeneic bone marrow transplantation. Acta Derm-Venereol. 2005;85:82–3.

    PubMed  Google Scholar 

  9. Fukumoto M, Murao K, Watanabe H, Kubo Y, Arase S. Generalized hypopigmented macules after bone-marrow transplantation. Clin Exp Dermatol. 2011;36:803–4.

    CAS  PubMed  Google Scholar 

  10. Hirokawa M, Kume M, Wu T, Ishikawa K, Sawada KI. Laryngeal stenosis associated with chronic graft-versus-host disease following unrelated bone marrow transplantation. Bone Marrow Transpl. 2006;38:387–8.

    CAS  Google Scholar 

  11. Jacobsohn DA, Ruble K, Moresi JM, Vogelsang GB. Rapid-onset leucodrema associated with graft-versus-host disease. Bone Marrow Transpl. 2002;30:705–6.

    CAS  Google Scholar 

  12. Jang S, Kim IS, Youn SW. Chronic graft-versus-host disease mimicking psoriasis in a patient with hemophagocytic lymphohistiocytosis. Ann Dermatol. 2016;28:90–3.

    PubMed  PubMed Central  Google Scholar 

  13. Kamińska EC, Larson RA, Petronic-Rosic V. Amelanocytic anhidrotic alopecia areata-like phenotype after allogeneic hematopoietic cell transplant. Arch Dermatol. 2012;148:931–4.

    PubMed  Google Scholar 

  14. Kang HY, Kang WH. Leukomelanoderma following acute cutaneous graft-versus-host disease. Eur J Dermatol. 2004;14:146–9.

    PubMed  Google Scholar 

  15. Lee HT, Chen WS, Chou CT, Chen MH, Tsai CY. Chronic graft-versus-host disease mimicking rapid progressive rheumatoid arthritis with atlantoaxial subluxation. BMJ Case Rep. 2009;2009:bcr06.2009.1941.

    PubMed  PubMed Central  Google Scholar 

  16. Martin-Gorgojo A, Martin JM, Gavrilova M, Monteagudo C, Jordá-Cuevas E. Chronic graft-versus-host disease presenting with coexisting diffuse melanoderma and hypopigmented patches: a peculiar presentation. Eur J Dermatol. 2013;23:553–5.

    PubMed  Google Scholar 

  17. Masunaga KTM, Kokuba H, Takahara M, Ohyama B, Hashimoto T, Furue M. Mucous membrane pemphigoid with antibodies to the b3 subunit of laminin 332. J Dermatol. 2011;38:1082–4.

    CAS  PubMed  Google Scholar 

  18. Mellouli F, Ksouri H, Dhouib N, Torjmen L, Abdelkefi A, Ladeb S, et al. Possible transfer of vitiligo by allogeneic bone marrow transplantation: a case report. Pediatr Transplant. 2009;13:1058–61.

    PubMed  Google Scholar 

  19. Miura T, Yamamoto T. Post-herpetic vitiligo in an 8-year-old boy with immunosuppression. J Dermatol. 2014;41:270–1.

    PubMed  Google Scholar 

  20. Miyawaki Y, Imoto I, Tokairin Y, Kawada K, Nakajima Y, Nishikage T, et al. Esophageal squamous cell carcinoma developed 11 years after allogeneic bone marrow transplantation for acute lymphatic leukemia. Jpn J Clin Oncol. 2013;43:69–73.

    PubMed  Google Scholar 

  21. Nagler A, Levi-Schaffer F, Bystryn JC, Klaus SN. Total leucoderma: a rare manifestation of cutaneous chronic graft-versus-host disease. Br J Dermatol. 1996;134:780–3.

    CAS  PubMed  Google Scholar 

  22. Nambudiri VE, Tsiaras WG, Schmidt BA, Huang JT. Total leukoderma and leukotrichia in a child after hematopoietic SCT: report of a case and review of the literature. Bone Marrow Transpl. 2014;49:460–2.

    CAS  Google Scholar 

  23. Neumeister P, Strunk D, Apfelbeck U, Sill H, Linkesch W. Adoptive transfer of vitiligo after allogeneic bone marrow transplantation for non-Hodgkin’s lymphoma. Lancet. 2000;355:1334–5.

    CAS  PubMed  Google Scholar 

  24. Oiso N, Kawada A. Vitiligo and remarkable freckles in chronic graft-versus-host disease. Eur J Dermatol. 2016;26:91–2.

    CAS  PubMed  Google Scholar 

  25. Sanli H, Akay BN, Arat M, Kocyigit P, Akan H, Beksac M, et al. Vitiligo after hematopoietic cell transplantation: six cases and review of the literature. Dermatology. 2008;216:349–54.

    PubMed  Google Scholar 

  26. Strouthidis NG, Francis PJ, Stanford MR, Graham EM, Holder GE, Bird AC. Posterior segment complications of graft versus host disease after bone marrow transplantation. Br J Ophthalmol. 2003;87:1421–3.

    CAS  PubMed  PubMed Central  Google Scholar 

  27. Tan AW, Koh LP, Goh BK. Leucoderma in chronic graft-versus-host disease: excellent repigmentation with noncultured cellular grafting. Br J Dermatol. 2011;165:435–7.

    CAS  PubMed  Google Scholar 

  28. Tang H, Wang C, Fu L, Xu AE. Successful repigmentation of vitiligo after allogeneic bone marrow transplantation for hodgkin’s lymphoma by autologous noncultured melanocyte-keratinocyte transplantation. Indian J Dermatol. 2015;60:470–2.

    PubMed  PubMed Central  Google Scholar 

  29. Williams JS, Mufti GJ, Du Vivier AWP, Salisbury JR, Creamer D. Leucoderma and leucotrichia in association with chronic cutaneous graft-versus-host disease. Br J Dermatol. 2008;158:172–4.

    CAS  PubMed  Google Scholar 

  30. Worswick S, Pirouz A, Lee DJ. Depigmented macules following autologous bone-marrow transplant. Arch Dermatol. 2011;147:1460–1.

    PubMed  Google Scholar 

  31. Chan CC, Yao M, Tsai TF. Diffuse depigmentation in a patient with chronic myeloid leukemia. J Am Acad Dermatol. 2006;54:738–40.

    PubMed  Google Scholar 

  32. Čeović R, Desnica L, Pulanić D, Serventi Seiwerth R, Ilić I, Grce M, et al. High frequency of cutaneous manifestations including vitiligo and alopecia areata in a prospective cohort of patients with chronic graft-vs-host disease. Croat Med J. 2016;57:229–38.

    PubMed  PubMed Central  Google Scholar 

  33. Khalil A, Zaidman I, Bergman R, Elhasid R, Ben-Arush MW. Autoimmune complications after hematopoietic stem cell transplantation in children with nonmalignant disorders. ScientificWorldJournal. 2014;2014:581657.

    PubMed  PubMed Central  Google Scholar 

  34. Kumar AJ, Hexner EO, Frey NV, Luger SM, Loren AW, Reshef R, et al. Pilot study of prophylactic ex vivo costimulated donor leukocyte infusion after reduced-intensity conditioned allogeneic stem cell transplantation. Biol Blood Marrow Transplant. 2013;19:1094–101.

    PubMed  Google Scholar 

  35. Lee PP, Chen TX, Jiang LP, Chen J, Chan KW, Lee TL, et al. Clinical and molecular characteristics of 35 Chinese children with Wiskott–Aldrich syndrome. J Clin Immunol. 2009;29:490–500.

    CAS  PubMed  Google Scholar 

  36. Marcus N, Takada H, Law J, Cowan MJ, Gil J, Regueiro JR, et al. Hematopoietic stem cell transplantation for CD3delta deficiency. J Allergy Clin Immunol. 2011;128:1050–7.

    CAS  PubMed  PubMed Central  Google Scholar 

  37. Straathof K, Anoop P, Allwood Z, Silva J, Nikolajeva O, Chiesa R, et al. Long-term outcome following cyclosporine-related neurotoxicity in paediatric allogeneic haematopoietic stem cell transplantation. Bone Marrow Transpl. 2017;52:159–62.

    CAS  Google Scholar 

  38. Zuo RC, Naik HB, Steinberg SM, Baird K, Mitchell SA, Kuzmina Z, et al. Risk factors and characterization of vitiligo and alopecia areata in patients with chronic graft-vs-host disease. JAMA Dermatol. 2015;15:23–32.

    Google Scholar 

  39. Claudy AL, Schmitt D, Freycon F, Boucheron S. Melanocyte-lymphocyte interaction in human graft-versus-host disease. J Cutan Pathol. 1983;10:305–11.

    CAS  PubMed  Google Scholar 

  40. Grimes PE. New insights and new therapies in vitiligo. J Am Med Assoc. 2005;293:730–5.

    CAS  Google Scholar 

  41. Mohammed GF, Gomaa AH, Al-Dhubaibi MS. Highlights in pathogenesis of vitiligo. World J Clin Cases. 2015;3:221–30.

    PubMed  PubMed Central  Google Scholar 

  42. Le Poole IC, van den Wijngaard RM, Westerhof W, Das PK. Presence of T cells and macrophages in inflammatory vitiligo skin parallels melanocyte disappearance. Am J Pathol. 1996;148:1219–28.

    PubMed  PubMed Central  Google Scholar 

  43. Ogg GS, Rod Dunbar P, Romero P, Chen JL, Cerundolo V. High frequency of skin-homing melanocyte-specific cytotoxic T lymphocytes in autoimmune vitiligo. J Exp Med. 1998;188:1203–8.

    CAS  PubMed  PubMed Central  Google Scholar 

  44. Lee SE, Cho BS, Kim JH, Yoon JH, Shin SH, Yahng SA, et al. Risk and prognostic factors for acute GVHD based on NIH consensus criteria. Bone Marrow Transpl. 2013;48:587–92.

    Google Scholar 

  45. Jagasia M, Arora M, Flowers ME, Chao NJ, McCarthy PL, Cutler CS, et al. Risk factors for acute GVHD and survival after hematopoietic cell transplantation. Blood. 2012;119:296–307.

    CAS  PubMed  PubMed Central  Google Scholar 

  46. Sherer Y, Shoenfeld Y. Autoimmune diseases and autoimmunity post-bone marrow transplantation. Bone Marrow Transpl. 1998;22:873–81.

    CAS  Google Scholar 

  47. Bakhtiar S, Gámez-Díaz L, Jarisch A, Soerensen J, Grimbacher B, Belohradsky B, et al. Treatment of infantile inflammatory bowel disease and autoimmunity by allogeneic stem cell transplantation in LPS-responsive beige-like anchor deficiency. Front Immunol. 2017;8:1–9.

    Google Scholar 

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  1. These authors contributed equally: Alessio Totani, Hossam Amin

Authors and Affiliations

  1. Faculty of Health and Medical Sciences, The University of Adelaide, Adelaide, SA, Australia

    Alessio Totani, Hossam Amin & Ian Lewis

  2. Royal Adelaide Hospital, Adelaide, SA, Australia

    Stephen Bacchi

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  1. Alessio Totani
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Correspondence to Alessio Totani.

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Totani, A., Amin, H., Bacchi, S. et al. Vitiligo following stem-cell transplant. Bone Marrow Transplant 55, 332–340 (2020). https://doi.org/10.1038/s41409-019-0626-x

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