Korean J Pediatr Hematol Oncol.  1998 Apr;5(1):207-215.

Bronchiolitis Obliterans and Generalized Vitiligo as Rare Presentations of Chronic Graft-versus-Host Disease

• Kim CJ
• Kook H
• Cho SH
• Choi SH
• Hwang MJ
• Hwang TJ

Abstract

Chronic graft-versus-host disease(GVHD), found in 30~50% of long-term survivors after an HLA-identical sibling transplant, in a multi-organ syndrome resembling collagen vascular diseases. Although the skin is the most commonly involved organ, generalized vitiligo with poliosis has rarely been reported as a manifestation. Patient 1 underwent an allogeneic peripheral blood stem cell transplant when late graft failure developed after initial bone marrow transplant(BMT) for aplastic anemia. She suffered from Grade III GVHD involving gut and skin, and developed cough, wheeze and dyspnea along with decreased pulmonary function. The diagnosis of bronchiolitis obliterans in association with chronic GVHD was made, and treatment with steroid and hydroxychloroquine(HCQ) resulted in some improvement. Patient 2 showed de novo chronic GVHD after allogeneic BMT for aplastic anemia. The vitiligo with premature grayness of hair and eyelash was the manifestation of chronic GVHD. Steroid and HCQ treatment stabilized the skin manifestations. HCQ treatment, though a larger study is needed, seemed to be safe adjuvant in the management of chronic GVHD in children.

Keyword

Chronic graft-versus-host disease; Bone marrow transplantation; Aplastic anemia; Bronchiolitis obliterans; Vitiligo; Steroid; Hydroxychloroquine

MeSH Terms

Anemia, Aplastic
Bone Marrow
Bone Marrow Transplantation
Bronchiolitis Obliterans*
Bronchiolitis*
Child
Collagen
Cough
Diagnosis
Dyspnea
Graft vs Host Disease*
Hair
Humans
Hydroxychloroquine
Siblings
Skin
Skin Manifestations
Stem Cells
Survivors
Transplants
Vascular Diseases
Vitiligo*
Collagen
Hydroxychloroquine