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Intracystic interferon-alpha in pediatric craniopharyngioma patients: an international multicenter assessment on behalf of SIOPE and ISPN.
Kilday, John-Paul; Caldarelli, Massimo; Massimi, Luca; Chen, Robert Hsin-Hung; Lee, Yi Yen; Liang, Muh-Lii; Parkes, Jeanette; Naiker, Thuran; van Veelen, Marie-Lise; Michiels, Erna; Mallucci, Conor; Pettorini, Benedetta; Meijer, Lisethe; Dorfer, Christian; Czech, Thomas; Diezi, Manuel; Schouten-van Meeteren, Antoinette Y N; Holm, Stefan; Gustavsson, Bengt; Benesch, Martin; Müller, Hermann L; Hoffmann, Anika; Rutkowski, Stefan; Flitsch, Joerg; Escherich, Gabriele; Grotzer, Michael; Spoudeas, Helen A; Azquikina, Kristian; Capra, Michael; Jiménez-Guerra, Rolando; MacDonald, Patrick; Johnston, Donna L; Dvir, Rina; Constantini, Shlomi; Kuo, Meng-Fai; Yang, Shih-Hung; Bartels, Ute.
Affiliation
  • Kilday JP; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Caldarelli M; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Massimi L; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Chen RH; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Lee YY; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Liang ML; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Parkes J; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Naiker T; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • van Veelen ML; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Michiels E; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Mallucci C; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Pettorini B; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Meijer L; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Dorfer C; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Czech T; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Diezi M; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Schouten-van Meeteren AYN; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Holm S; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Gustavsson B; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Benesch M; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Müller HL; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Hoffmann A; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Rutkowski S; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Flitsch J; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Escherich G; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Grotzer M; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Spoudeas HA; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Azquikina K; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Capra M; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Jiménez-Guerra R; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • MacDonald P; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Johnston DL; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Dvir R; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Constantini S; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Kuo MF; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Yang SH; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
  • Bartels U; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Oxford Road, Manchester, England, UK; The Centre for Paediatric, Teenage and Young Adult Cancer, Institute of Cancer Sciences, The University of Manchester, England, UK; Department of Pediatric Neurosurgery, A Ge
Neuro Oncol ; 19(10): 1398-1407, 2017 Oct 01.
Article in En | MEDLINE | ID: mdl-28499018
ABSTRACT

BACKGROUND:

Craniopharyngiomas are frequent hypothalamo-pituitary tumors in children, presenting predominantly as cystic lesions. Morbidity from conventional treatment has focused attention on intracystic drug delivery, hypothesized to cause fewer clinical consequences. However, the efficacy of intracystic therapy remains unclear. We report the retrospective experiences of several global centers using intracystic interferon-alpha.

METHODS:

European Société Internationale d'Oncologie Pédiatrique and International Society for Pediatric Neurosurgery centers were contacted to submit a datasheet capturing pediatric patients with cystic craniopharyngiomas who had received intracystic interferon-alpha. Patient demographics, administration schedules, adverse events, and outcomes were obtained. Progression was clinical or radiological (cyst reaccumulation, novel cysts, or solid growth).

RESULTS:

Fifty-six children (median age, 6.3 y) from 21 international centers were identified. Median follow-up from diagnosis was 5.1 years (0.3-17.7 y). Lesions were cystic (n = 22; 39%) or cystic/solid (n = 34; 61%). Previous progression was treated in 43 (77%) patients before interferon use. In such cases, further progression was delayed by intracystic interferon compared with the preceding therapy for cystic lesions (P = 0.0005). Few significant attributable side effects were reported. Progression post interferon occurred in 42 patients (median 14 mo; 0-8 y), while the estimated median time to definitive therapy post interferon was 5.8 (1.8-9.7) years.

CONCLUSIONS:

Intracystic interferon-alpha can delay disease progression and potentially offer a protracted time to definitive surgery or radiotherapy in pediatric cystic craniopharyngioma, yet demonstrates a favorable toxicity profile compared with other therapeutic modalities-important factors for this developing age group. A prospective, randomized international clinical trial assessment is warranted.
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Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Pituitary Neoplasms / Interferon-alpha / Craniopharyngioma Type of study: Observational_studies / Prognostic_studies Limits: Adolescent / Child / Child, preschool / Female / Humans / Male Language: En Journal: Neuro Oncol Journal subject: NEOPLASIAS / NEUROLOGIA Year: 2017 Type: Article
Fulltext
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Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Pituitary Neoplasms / Interferon-alpha / Craniopharyngioma Type of study: Observational_studies / Prognostic_studies Limits: Adolescent / Child / Child, preschool / Female / Humans / Male Language: En Journal: Neuro Oncol Journal subject: NEOPLASIAS / NEUROLOGIA Year: 2017 Type: Article