Intracystic interferon-alpha in pediatric craniopharyngioma patients: an international multicenter assessment on behalf of SIOPE and ISPN

被引：64

作者：

Kilday, John-Paul ^{[1
,2
]}

Caldarelli, Massimo ^{[3
]}

Massimi, Luca ^{[3
]}

Chen, Robert Hsin-Hung ^{[4
]}

Lee, Yi Yen ^{[4
]}

Liang, Muh-Lii ^{[4
]}

Parkes, Jeanette ^{[5
]}

Naiker, Thuran ^{[5
]}

van Veelen, Marie-Lise ^{[6
]}

Michiels, Erna ^{[6
]}

Mallucci, Conor ^{[7
]}

Pettorini, Benedetta ^{[7
]}

Meijer, Lisethe ^{[8
]}

Dorfer, Christian ^{[9
]}

Czech, Thomas ^{[9
]}

Diezi, Manuel ^{[10
]}

Schouten-van Meeteren, Antoinette Y. N. ^{[11
]}

Holm, Stefan ^{[12
]}

Gustavsson, Bengt ^{[12
]}

Benesch, Martin ^{[13
]}

Mueller, Hermann L. ^{[14
]}

Hoffmann, Anika ^{[14
]}

Rutkowski, Stefan ^{[15
,16
]}

Flitsch, Joerg ^{[15
,16
]}

Escherich, Gabriele ^{[15
,16
]}

Grotzer, Michael ^{[17
]}

Spoudeas, Helen A. ^{[18
,19
]}

Azquikina, Kristian ^{[18
,19
]}

Capra, Michael ^{[20
]}

Jimenez-Guerra, Rolando ^{[21
]}

MacDonald, Patrick ^{[22
]}

Johnston, Donna L. ^{[23
]}

Dvir, Rina ^{[24
]}

Constantini, Shlomi ^{[24
]}

Kuo, Meng-Fai ^{[25
]}

Yang, Shih-Hung ^{[25
]}

Bartels, Ute ^{[26
]}

机构：

[1] Royal Manchester Childrens Hosp, Childrens Brain Tumour Res Network, Oxford Rd, Manchester, Lancs, England

[2] Univ Manchester, Inst Canc Sci, Ctr Paediat Teenage & Young Adult Canc, Manchester, Lancs, England

[3] A Gemelli Hosp, Dept Pediat Neurosurg, Rome, Italy

[4] Taipei Vet Gen Hosp, Neurol Inst, Div Pediat Neurosurg, Taipei, Taiwan

[5] Univ Cape Town, Groote Schuur Hosp, Cape Town, South Africa

[6] Sophia Childrens Univ Hosp, Erasmus MC, Rotterdam, Netherlands

[7] Alder Hey Childrens Hosp NHS Fdn Trust, Dept Neurosurg, Liverpool, Merseyside, England

[8] Univ Groningen, Univ Med Ctr Groningen, Beatrix Childrens Hosp, Groningen, Netherlands

[9] Med Univ Vienna, Dept Neurosurg, Vienna, Austria

[10] Univ Hosp Lausanne, Pediat Haematooncol Unit, Lausanne, Switzerland

[11] Acad Med Ctr, Emma Childrens Hosp, Dept Pediat Oncol, Amsterdam, Netherlands

[12] Karolinska Univ Hosp, Dept Pediat Neurosurg, Stockholm, Sweden

[13] Med Univ Graz, Dept Pediat & Adolescent Med, Div Pediat Hematol & Oncol, Graz, Austria

[14] Med Campus Univ Oldenburg, Klinikum Oldenburg AoR, Dept Pediat, Oldenburg, Germany

[15] Univ Med Ctr Hamburg Eppendorf, Dept Neurosurg, Hamburg, Germany

[16] Univ Med Ctr Hamburg Eppendorf, Dept Oncol, Hamburg, Germany

[17] Univ Childrens Hosp Zurich, Dept Neurosurg, Zurich, Switzerland

[18] Great Ormond St Hosp Sick Children, Dept Paediat Neuroendocrinol, London, England

[19] Great Ormond St Hosp Sick Children, Dept Neurosurg, London, England

[20] Our Ladys Childrens Hosp, Dept Paediat, Crumlin, Ireland

[21] Hosp Angeles Mexico, Dept Pediat Neurosurg, Mexico City, DF, Mexico

[22] Winnipeg Childrens Hosp, Dept Neurosurg, Winnipeg, MB, Canada

[23] Childrens Hosp Eastern Ontario, Dept Paediat, Ottawa, ON, Canada

[24] Tel Aviv Sourasky Med Ctr, Dept Neurosurg, Tel Aviv, Israel

[25] Natl Taiwan Univ Hosp, Dept Neurosurg, Taipei, Taiwan

[26] Hosp Sick Children, Dept Paediat Oncol, Toronto, ON, Canada

来源：

NEURO-ONCOLOGY | 2017年 / 19卷 / 10期

关键词：

craniopharyngioma; intracystic interferon; pediatric; retrospective; PHASE-II TRIAL; SQUAMOUS-CELL CARCINOMA; CYSTIC-CRANIOPHARYNGIOMA; CHILDHOOD CRANIOPHARYNGIOMA; INTRALESIONAL INTERFERON-ALPHA-2B; INTRATUMORAL CHEMOTHERAPY; INTRACAVITARY IRRADIATION; RADIATION-THERAPY; FOLLOW-UP; MANAGEMENT;

D O I：

10.1093/neuonc/nox056

中图分类号：

R73 [肿瘤学];

学科分类号：

100214 ;

摘要：

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 Societe Internationale d'Oncologie Pediatrique 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.

引用

页码：1398 / 1407

页数：10

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