Use of 2-chlorodeoxyadenosine to treat infantile myofibromatosis.
A 3-year-old boy had fever and bone pain. Magnetic resonance imaging of his femurs showed marrow replacement; iliac crest marrow biopsy revealed myelofibrosis. Although the pathologic criteria for Langerhans cell histiocytosis were not met, the clinical picture led to treatment with etoposide and methylprednisolone, without clinical improvement. One month after presentation, generalized tonic-clonic seizures occurred, and magnetic resonance imaging revealed parenchymal brain lesions. 2-chlorodeoxyadenosine was used. Because of the unexpected lack of response to etoposide and methylprednisolone, a second bone biopsy was performed. The diagnosis was revised to infantile myofibromatosis. After six courses of 2-chlorodeoxyadenosine, brain and bone lesions regressed, with resolution of the clinical symptoms.
Williams
Wanika
W
Baylor College of Medicine, Houston, Texas, USA.
Craver
Randall D
RD
Correa
Hernan
H
Velez
Maria
M
Gardner
Renee V
RV
D000970:Antineoplastic Agents / Q000627:therapeutic use*; D001853:Bone Marrow / Q000473:pathology; D002675:Child, Preschool; D017338:Cladribine / Q000627:therapeutic use*; D004830:Epilepsy, Tonic-Clonic / Q000209:etiology; D006801:Humans; D008279:Magnetic Resonance Imaging; D008297:Male; D018224:Myofibromatosis / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy* / Q000473:pathology; D016896:Treatment Outcome
D000970:Antineoplastic Agents; D017338:Cladribine