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Rapid-onset leucodrema associated with graft-versus-host disease

Bone Marrow Transplantation volume 30, pages 705–706 (2002)Cite this article

Chronic graft-versus-host disease (cGVHD) is a major complication of allogeneic hematopoietic cell transplantation (HCT) that occurs in 25–75% of patients, depending on the source of the graft.1 The typical skin lesions are either lichenoid or sclerodermatous. Patients may also have areas of leucoderma, alopecia, bullae and photosensitivity.2,3,4 While partial leucoderma (hypopigmentation) occurs frequently, total body leucoderma is extremely unusual in cGVHD. One case report in the literature describes a 14-year-old boy in whom total leucoderma of the skin occurred gradually over a few months. This patient had an elevated level of anti-melanocyte antibodies in his serum, which may be responsible for the leucoderma.5 We describe a patient who developed total leucoderma arising from his cGVHD. In contrast to the previously described case, our patient developed total leucoderma very acutely, over the course of 2 weeks.

A 14-year-old white male underwent a matched-unrelated donor bone marrow transplant in 1987 for chronic myelogenous leukemia. His preparative regimen consisted of busulfan, cytosine arabinoside (Ara-C) and total body irradiation (TBI). The donor marrow was T cell-depleted and he received cyclosporine and steroids as GVHD prophylaxis. He had no significant acute GVHD. However, upon tapering his prophylaxis he experienced chronic GVHD. He had multiple episodes of worsening of his chronic GVHD on tapering of his immunosuppressive medications. These episodes included widespread erythema with lichenoid features, scaling and alopecia. A skin biopsy 5 months after transplant showed acanthosis, exocytosis of mononuclear cells, and a perivascular mononuclear cell infiltrate. The patient responded promptly to higher doses of steroids. After the third attempt to wean his steroid therapy the patient experienced a complete loss of pigment. This phenomenon was first noted when his hair grew in white and was followed by a splotchy depigmentation of the skin. Within 2 weeks he was completely depigmented. There was no other organ system involved. The patient remained on cyclosporine and steroids for more than a year before he tolerated a steroid withdrawal.

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Authors and Affiliations

  1. Oncology and Pediatrics, The Johns Hopkins University School of Medicine, Baltimore, MD, USA

    DA Jacobsohn & K Ruble

  2. Dermatology and Pathology, The Johns Hopkins University School of Medicine, Baltimore, MD, USA

    JM Moresi

  3. Department of Oncology, Hematologic Malignancies, The Johns Hopkins University School of Medicine, Baltimore, MD, USA

    GB Vogelsang

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  1. DA Jacobsohn
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  2. K Ruble
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  3. JM Moresi
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  4. GB Vogelsang
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Jacobsohn, D., Ruble, K., Moresi, J. et al. Rapid-onset leucodrema associated with graft-versus-host disease. Bone Marrow Transplant 30, 705–706 (2002). https://doi.org/10.1038/sj.bmt.1703724

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