title: Intracystic bleomycin therapy for craniopharyngioma in children: the Canadian experience.
abstract: BACKGROUND
  Surgical removal and radiation therapy are associated with significant risk for morbidity in the pediatric population with craniopharyngioma. Intracystic therapies have been utilized in some centers to potentially decrease morbidity associated with cystic craniopharyngioma. The aim of the study was to review the Canadian experience with intracystic bleomycin therapy (ICB).
  
  
  METHODS
  All centers in the Canadian Pediatric Brain Tumor Consortium (CPBTC) were invited to participate in a retrospective review of this treatment. A questionnaire was sent to each center. The data were analyzed at British Columbia's Children's Hospital.
  
  
  RESULTS
  In all, 6 of 17 centers utilized ICB and submitted data. A total of 17 of 19 patients with the intention to treat received bleomycin. Twelve were treated at the time of diagnosis, and 5 at the time of recurrence. Five patients achieved a complete response, 6 achieved a partial response, and 5 achieved a minor response to bleomycin. One patient was stable for 2.8 years. At the time of last follow-up, 8 patients have not required further intervention. Complications included transient symptomatic peritumoral edema (2 patients), precocious puberty (1 patient) and panhypopituitarism (2 patients). The median follow-up was 4 years (range, 0.5-10.2 years). The median progression-free survival was 1.8 years (range, 0.3-6.1 years). One patient died of a massive infarct secondary to radiation-induced moyamoya syndrome.
  
  
  CONCLUSIONS
  ICB was found to be well tolerated in this group of children. ICB may be a feasible and effective therapy for certain children with craniopharyngioma. Bleomycin may delay the need for aggressive surgery or radiation therapy for several years. Prospective multiinstitutional clinical trials are required to further evaluate the feasibility, effectiveness, and dose schedules of this treatment.
authors.affiliation: Division of Neurology and Oncology, British Columbia's Children's Hospital, Vancouver, British Columbia, Canada. jhukin@cw.bc.ca
authors.forename: Juliette
authors.initials: J
authors.lastname: Hukin
authors.forename: Paul
authors.initials: P
authors.lastname: Steinbok
authors.forename: Lucie
authors.initials: L
authors.lastname: Lafay-Cousin
authors.forename: Glenda
authors.initials: G
authors.lastname: Hendson
authors.forename: Douglas
authors.initials: D
authors.lastname: Strother
authors.forename: Claude
authors.initials: C
authors.lastname: Mercier
authors.forename: Yvan
authors.initials: Y
authors.lastname: Samson
authors.forename: William
authors.initials: W
authors.lastname: Howes
authors.forename: Eric
authors.initials: E
authors.lastname: Bouffet
mesh_terms: D000293:Adolescent; D000903:Antibiotics, Antineoplastic / Q000008:administration & dosage* / Q000009:adverse effects; D001761:Bleomycin / Q000008:administration & dosage* / Q000009:adverse effects; D002648:Child; D002675:Child, Preschool; D003397:Craniopharyngioma / Q000000981:diagnostic imaging / Q000188:drug therapy*; D018572:Disease-Free Survival; D005260:Female; D006801:Humans; D007223:Infant; D015552:Injections, Intralesional; D008297:Male; D010911:Pituitary Neoplasms / Q000000981:diagnostic imaging / Q000188:drug therapy*; D011859:Radiography
chemical_list: D000903:Antibiotics, Antineoplastic; D001761:Bleomycin