Isolated juvenile xanthogranuloma in the bone marrow: report of a case and review of the literature.
We report a case of juvenile xanthogranuloma limited to involvement of the bone marrow in a 6-week-old male infant. Evaluation of the bone marrow was a part of the workup for peripheral blood cytopenia. Examination showed hypercellular marrow with paratrabecular clusters of lipidized histiocytes positive for CD68, CD4, and factor XIII(a) and negative for S100 and CD1a. Clinical and radiological workup showed no associated skin lesions or osseous or visceral involvement. The patient was started on chemotherapy with clinical improvement and gradual decreased bone marrow involvement. The child is alive and well at 16 months of age. This case represents, to the best of our knowledge, the 1st documented case of juvenile xanthogranuloma with isolated bone marrow involvement sparing skin and viscera.
Kesserwan
Chimen
C
Department of Laboratory Medicine, Children's Hospital, 700 Children's Drive, Columbus, OH 43205, USA.
Boué
Daniel R
DR
Kahwash
Samir B
SB
D015703:Antigens, CD / Q000378:metabolism; D000972:Antineoplastic Agents, Phytogenic / Q000627:therapeutic use; D001706:Biopsy; D001853:Bone Marrow / Q000187:drug effects / Q000378:metabolism / Q000473:pathology* / Q000601:surgery; D026122:Factor XIIIa / Q000378:metabolism; D005500:Follow-Up Studies; D006801:Humans; D007150:Immunohistochemistry; D007223:Infant; D018950:Lipopolysaccharide Receptors / Q000378:metabolism; D008297:Male; D011859:Radiography; D017463:Receptors, Complement 3b / Q000378:metabolism; D013997:Time Factors; D016896:Treatment Outcome; D014746:Vimentin / Q000378:metabolism; D014750:Vincristine / Q000627:therapeutic use; D014972:Xanthogranuloma, Juvenile / Q000000981:diagnostic imaging / Q000188:drug therapy / Q000378:metabolism / Q000473:pathology*
D015703:Antigens, CD; D000972:Antineoplastic Agents, Phytogenic; D018950:Lipopolysaccharide Receptors; D017463:Receptors, Complement 3b; D014746:Vimentin; D014750:Vincristine; D026122:Factor XIIIa