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Systemic Juvenile Xanthogranuloma Involving the Bone Marrow, Multiple Bones, and the Skin That Developed During Treatment of Acute Lymphoblastic Leukemia in Remission State

Authors
Cheon, E; Yang, S; Han, JH; Lee, KC; Park, JE
Citation
Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society, 21(5):489-493, 2018
Journal Title
Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society
ISSN
1093-52661615-5742
Abstract
Juvenile xanthogranuloma (JXG) is a rare benign disorder classified as non-Langerhans cell histiocytosis, with unclear etiology and pathogenesis. JXG is generally characterized by solitary or multiple cutaneous nodules that resolve spontaneously over a few years. JXG rarely presents as extracutaneous lesions that progress to a symptomatic systemic disorder through multiple organ involvement. We encountered a systemic JXG case involving the bone marrow, multiple bones, and the skin during acute lymphoblastic leukemia (ALL) treatment. A 16-year-old boy undergoing ALL treatment experienced unexplained prolonged fever and scalp, hip joint, and right knee joint pain for 2 weeks during interim maintenance chemotherapy. Bone marrow pathologic findings revealed no evidence of leukemia relapse but showed diffuse infiltration of histiocytes with several Touton-type giant cells: the stains were positive for CD68 and negative for CD1a and S100 protein. Bone and skin biopsies confirmed the findings. Symptoms have resolved since maintenance chemotherapy, which included vincristine, dexamethasone, 6-mercaptopurine, and methotrexate. Bone marrow involvement of JXG is very rare, occurring only in patients less than 1 year of age: however, this case was reported in an adolescent during ALL treatment.
Keywords
juvenile xanthogranulomasystemicbone marrowlymphoblastic leukemia
MeSH terms
AdolescentAntineoplastic Agents/therapeutic useBone Marrow/pathology*Bone and Bones/pathology*HumansMalePrecursor Cell Lymphoblastic Leukemia-Lymphoma/complications*Precursor Cell Lymphoblastic Leukemia-Lymphoma/drug therapyPrecursor Cell Lymphoblastic Leukemia-Lymphoma/pathologySkin/pathology*Xanthogranuloma, Juvenile/complications*Xanthogranuloma, Juvenile/pathology
DOI
10.1177/1093526617721775
PMID
28836893
Appears in Collections:
Journal Papers > School of Medicine / Graduate School of Medicine > Pathology
Journal Papers > School of Medicine / Graduate School of Medicine > Pediatrics & Adolescent Medicine
AJOU Authors
한, 재호박, 준은
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