title: Premature physeal closure following 13-cis-retinoic acid and prolonged fenretinide administration in neuroblastoma.
abstract: Retinoid therapy has contributed to improved outcomes in neuroblastoma. Clinical trials of fenretinide report favorable toxicity and disease stabilization in patients with high risk (HR) neuroblastoma. Skeletal effects have been described with other retinoids, but not with fenretinide to date. Two patients with HR, metastatic, refractory neuroblastoma received protracted courses of oral fenretinide for more than 5 years' duration. Both developed premature long bone physeal closure, causing limb length discrepancies; their neuroblastoma remains in remission. The radiographic and clinical findings reported suggest these skeletal abnormalities may be a consequence of treatment with 13-cis-retinoic acid (13cisRA) followed by prolonged oral fenretinide exposure.
authors.affiliation: Department of Pediatrics, Stanford University School of Medicine, Stanford, California. asteine@stanford.edu.
authors.forename: Angela
authors.initials: A
authors.lastname: Steineck
authors.affiliation: Department of Radiology, University of California at San Francisco, San Francisco, California.
authors.forename: John D
authors.initials: JD
authors.lastname: MacKenzie
authors.affiliation: Department of Pediatrics, Division of Hematology/Oncology, Stanford University School of Medicine, Stanford, California.
authors.forename: Clare J
authors.initials: CJ
authors.lastname: Twist
mesh_terms: D000970:Antineoplastic Agents / Q000009:adverse effects*; D002648:Child; D002675:Child, Preschool; D005260:Female; D017313:Fenretinide / Q000009:adverse effects*; D006132:Growth Plate / Q000187:drug effects*; D006801:Humans; D015474:Isotretinoin / Q000009:adverse effects*; D008297:Male; D009447:Neuroblastoma / Q000188:drug therapy*
chemical_list: D000970:Antineoplastic Agents; D017313:Fenretinide; D015474:Isotretinoin
keywords: fenretinide; late-effects; neuroblastoma; pediatric oncology; premature physeal closure; skeletal toxicity