@article{cfc11d6dbc9d4ec9871f874ece8f950d,
title = "Intracystic interferon-alpha in pediatric craniopharyngioma patients: An international multicenter assessment on behalf of SIOPE and ISPN",
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{\'e}t{\'e} Internationale d'Oncologie P{\'e}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.",
keywords = "Adolescent, Cancer Research, Child, Child, Preschool, Craniopharyngioma, Female, Humans, Injections, Intralesional, Interferon-alpha, Intracystic interferon, Male, Neurology (clinical), Oncology, Pediatric, Pituitary Neoplasms, Retrospective, Retrospective Studies, Adolescent, Cancer Research, Child, Child, Preschool, Craniopharyngioma, Female, Humans, Injections, Intralesional, Interferon-alpha, Intracystic interferon, Male, Neurology (clinical), Oncology, Pediatric, Pituitary Neoplasms, Retrospective, Retrospective Studies",
author = "John-Paul Kilday and Massimo Caldarelli and Luca Massimi and Chen, {Robert Hsin-Hung} and Lee, {Yi Yen} and Muh-Lii Liang and Jeanette Parkes and Thuran Naiker and {Van Veelen}, Marie-Lise and Erna Michiels and Conor Mallucci and Benedetta Pettorini and Pettorini, {Benedetta Ludovica} and Lisethe Meijer and Christian Dorfer and Thomas Czech and Manuel Diezi and {Schouten-Van Meeteren}, {Antoinette Yn} and Stefan Holm and Bengt Gustavsson and Martin Benesch and M{\"u}ller, {Hermann L.} and Anika Hoffmann and Stefan Rutkowski and Joerg Flitsch and Gabriele Escherich and Michael Grotzer and Spoudeas, {Helen A.} and Kristian Azquikina and Michael Capra and Jimcrossed, {Signnez-Guerra Rolando} and {Mac Donald}, Patrick and Johnston, {Donna L.} and Rina Dvir and Shlomi Constantini and Meng-Fai Kuo and Shih-Hung Yang and Ute Bartels",
year = "2017",
doi = "10.1093/neuonc/nox056",
language = "English",
volume = "19",
pages = "1398--1407",
journal = "Neuro-Oncology",
issn = "1522-8517",
publisher = "Oxford University Press",
}