Intracystic interferon-alpha in pediatric craniopharyngioma patients: An international multicenter assessment on behalf of SIOPE and ISPN
Journal
Neuro-Oncology
Journal Volume
19
Journal Issue
10
Pages
1398-1407
Date Issued
2017
Author(s)
Kilday J.-P.
Caldarelli M.
Massimi L.
Chen R.H.-H.
Lee Y.Y.
Liang M.-L.
Parkes J.
Naiker T.
Van Veelen M.-L.
Michiels E.
Mallucci C.
Pettorini B.
Meijer L.
Dorfer C.
Czech T.
Diezi M.
Schouten-Van Meeteren A.Y.
Holm S.
Gustavsson B.
Benesch M.
M?ller H.L.
Hoffmann A.
Rutkowski S.
Flitsch J.
Escherich G.
Grotzer M.
Spoudeas H.A.
Azquikina K.
Capra M.
JimCrossed S.G.R.
Mac Donald P.
Johnston D.L.
Dvir R.
Constantini S.
Bartels U.
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 Soci?t? Internationale d'Oncologie P?diatrique 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. ? 2017 The Author(s).
SDGs
Other Subjects
alpha interferon; radioisotope; steroid; alpha interferon; acute disease; adolescent; Article; brain atrophy; brain edema; cancer control; child; clinical evaluation; clinical outcome; cohort analysis; conformal radiotherapy; confusion; controlled study; craniopharyngioma; demography; disease exacerbation; drug efficacy; drug safety; endocrine surgery; fatigue; female; gamma knife radiosurgery; headache; human; human tissue; hydrocephalus; hypernatremia; hyponatremia; incontinence; influenza; international cooperation; loss of appetite; major clinical study; malaise; male; medical society; multicenter study (topic); multiple cycle treatment; neurosurgery; preschool child; proton therapy; radiodiagnosis; retrospective study; school child; side effect; treatment response; tumor growth; visual disorder; weight reduction; clinical trial; craniopharyngioma; hypophysis tumor; intralesional drug administration; metabolism; multicenter study; procedures; Adolescent; Child; Child, Preschool; Craniopharyngioma; Female; Humans; Injections, Intralesional; Interferon-alpha; Male; Pituitary Neoplasms; Retrospective Studies
Publisher
Oxford University Press
Type
journal article