Interferon-alfa in the management of cystic craniopharyngioma in children under 5 years of age (original) (raw)
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Neuro-oncology, 2017
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. 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). Fifty-six children (median age, 6.3 y) from 21 international centers were identified. Median follow-up from diagnosis was 5...
International Journal of Case Reports and Images, 2013
Introduction: the use of intracystic treatments is relatively new in the treatment of craniopharyngioma and has the potential for decreased morbidity and long-term control in patients with recurrent cysts after radiotherapy as well as delaying radiotherapy treatment in young children. We describe a case in which the therapeutic use of interferon alpha-2b (IFNa-2b) was associated with an adverse effect that has not yet been documented in the medical literature. case report: An eight-year-old boy with a history of a craniopharyngioma was presented with radiological evidence of recurrence. He was first diagnosed at the age of eight months and had undergone repeated subtotal resections. At the age of three, he also received adjuvant radiotherapy after one of these resections. On this presentation of recurrence, a decision was made to offer him intracystic treatment with IFNa-2b. Although the typical pre-treatment leakage test was positive, the planned treatment decision was maintained following extensive multi-disciplinary consult with both local and international teams who advised that any
Neurosurgical Focus, 2010
C raniopharyngiomas are benign slow-growing lesions that challenge every neurosurgeon, even since the advent of major technological developments, with important refinements, such as microscopic techniques, ultrasonic aspirators, neuroendoscopes, and hormone replacement therapy. In major centers of pediatric neurosurgery with excellent endocrinological support, is not uncommon to see patients with CPs suffer deterioration with the onset of obesity, panhypopituitarism, hypercholesterolemia, and psychological disorders. 9 If on the one hand a complete resection of these lesions is tremendously satisfying to neurosurgeons, on the other hand this feeling is temporary because the lesion and the effects of hypothalamic dysfunction will recur. The use of intratumoral chemotherapy with the cytokine INFα is a simple method, with a very low cost, that allows the control of these tumors.
Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2017
Management of craniopharyngioma in children is challenging, and their quality of life can be significantly affected. Series describing this from low-middle income countries (LMIC) are few. The study provides a retrospective chart review of pediatric patients <18 years old, diagnosed with craniopharyngioma between 2003 and 2014, and treated at King Hussein Cancer Center, Jordan. Twenty-four patients (12 males) were identified. Median age at diagnosis was 7.4 years (0.9-16.4 years). Commonest symptoms were visual impairment and headache (71%). Review of seventeen preoperative MRIs showed hypothalamic involvement in 88% and hydrocephalus in 76%. Thirteen patients (54%) had multiple surgical interventions. Five patients (21%) had initial gross total resection. Eleven patients (46%) received radiotherapy and six (25%) intra-cystic interferon. Five years' survival was 87 ± 7% with a median follow-up of 4.5 years (0.3-12.3 years). Four patients (17%) died; one after post-operative c...
20-Year experience in childhood craniopharyngioma
International Journal of Radiation Oncology*Biology*Physics, 1993
Purpose: The management of craniopharyngioma is controversial, and surgery alone is frequently advocated. The purpose of this study was to assess the long-term impact of various treatments in childhood craniopharyngioma. Methods and Materials: Sixty-one children I 21 years of age at diagnosis were treated for craniopharyngioma at Children's Hospital and the Joint Center for Radiation Therapy in Boston from 1970 to 1990. The median age was 7.5 years (range 10 months-21 years). There were 33 females and 28 males. The median follow-up was 10 years (range 2-20.5 years). Neuroimaging was available for detailed review in 53. Nine children were treated with radiotherapy alone, 15 were treated with surgery alone, and 37 were treated with both surgery and radiotherapy. All patients in the radiotherapy and surgery plus radiotherapy groups were treated with megavoltage radiaiion with a median dose of 5464 cGy. Results: All nine of the children treated with radiation therapy alone are alive; none have recurred. Nine of the 15 children treated with surgery alone have recurred (p = 0.007 Fisher exact test). Two are alive with disease, and seven are alive without disease after treatment at relapse with radiation therapy, surgery, or both. Seven of the 37 patients treated with surgery plus radiotherapy have recurred. Three of the seven patients are dead of disease, three patients are alive with disease, and one patient is alive without disease after further treatment. The IO-year actuarial overall survival was 91% for al1 patients. The IO-year actuarial freedom from progression for the surgery group was 31% compared with 100% for patients treated with radiation therapy only (log rank p = O.Ol), and 86% for patients treated with surgery plus radiotherapy at diagnosis (p = 0.001). There were two treatment related deaths, both in the surgery plus radiotherapy group. A higher incidence of visual loss and diabetes insipidus was associated with the use of aggressive surgery. The size of the tumor at presentation correlated with an increased risk of recurrence; 5 of 6 patients with tumors 2 5 cm experienced recurrences while only 6 of 30 recurred when the tumor was < 5 cm. Conclusions: Overall survival in childhood craniopharyngioma is excellent. However, patients treated with surgery alone have a significantly worse freedom from progression when compared to patients treated with surgery and radiation therapy or radiation therapy alone.
Treatment outcomes of pediatric craniopharyngioma : a 15-year retrospective review of 35 cases
Journal of Korean Neurosurgical Society, 2012
The aim of this study was to describe a single center's experience in the management of craniopharyngiomas in children over a 15-year period. The clinical records of pediatric patients treated for craniopharyngiomas between December 1995 and February 2011 were reviewed. Thirty-five pediatric patients diagnosed with craniopharyngioma were treated, and their medical records and imaging data were analyzed retrospectively. The mean follow-up duration was 76 months (range, 10-195). Overall survival and local control rates at 10 years were 94.7±5.1% and 37.1±11.9%, respectively. The female-to-male ratio was 16 : 19, and the mean age was 8.6 years (range, 1-17). Initially, gross total resection (GTR) was performed in 30 patients; subtotal resection (STR) followed by radiotherapy was performed in 5 patients. Of the 14 cases that showed recurrence after GTR, 5 patients were treated with GTR, 1 with radiation therapy (RT), 4 with gamma knife radiosurgery (GKRS), and 4 with subtotal resect...
Clinical and Laboratory Follow up of Pediatric Craniopharyngioma Cases
Güncel Pediatri, 2020
2 , Betül Sevinir (0000-0002-3232-7652) 3 , M. Özgür Taşkapılıoğlu (0000-0001-5472-9065) 4 , Ömer Tarım (0000-0002-5322-5508) 2 ÖZ GİRİŞ ve AMAÇ: Bu çalışmada çocukluk çağı kraniyofaringioma olgularının klinik ve laboratuar bulguları ve izlem süresince karşılaşılan endokrin sorunların değerlendirilmesi amaçlanmıştır. YÖNTEM ve GEREÇLER: Çalışmaya Ocak 2010-Aralık 2017 arasında kraniyofaringiyoma tanısı alan olgular dahil edildi. Demografik ve klinik veriler Uludağ Üniversitesi Tıp Fakültesi Çocuk Endokrinoloji poliklinik dosyalarından retrospektif olarak değerlendirildi. İstatistiksel analizler için SPSS software version 21 kullanıldı. BULGULAR: Sekiz yıllık süreçte tanı alan 28 olgu çalışmaya dahil edildi. Olguların yaşları 60-207 ay arasında değişmekte olup median yaş 138,5 aydı. Cinsiyet dağılımlarına bakıldığında %53 erkek(n=15) ve %47 kızdı (n=13). En sık başvuru bulguları sırayla başağrısı, görme bozuklukları ve boy kısalığıydı. Tanı anında olguların %71'inde (n: 20) tümör boyutu 3 cm'den büyüktü. Preoperatif dönemde büyüme hormonu eksikliği %17(n=5) ve hipotiroidizm %14(n=4), hipokortizolemi %10 (n=3), diyabet insipit %7,1(n=2) sıklıkta saptandı. Postoperatif dönemde olguların %89,3'ünde çoklu hipofizer hormon eksikliği saptandı. TARTIŞMA ve SONUÇ: Kraniyofaringiyomalar yavaş büyüyen tümörler olup genellikle geç tanı alır. Geç tanı ve tümör boyutunun büyük olması sıklıkla endokrin kayıpların daha ciddi olmasına yol açar. Büyümede duraksama ve ilerleyici kilo artışı hipofizer kitlelerin erken tanısı açısından uyarıcı bulgulardır.
Child's Nervous System, 2019
Purpose To demonstrate the paradigm shift in management strategies of pediatric craniopharyngioma at our institution over the past six decades. Methods Retrospective analysis of all pediatric patients with craniopharyngioma treated at Boston Children's Hospital between 1960 and 2017. Results One hundred seventy-eight patients with craniopharyngioma were treated between 1960 and 2017; 135 (70 males and 65 females) fulfilled the inclusion criteria. Forty-five patients were treated in the old era (1960-1984) and 90 patients were treated in the new era (1985-2017). Gross total resection (GTR) was achieved in 4% and 43% of patients in old and new eras respectively. Sub-total resection (STR) and radiotherapy (XRT) were performed in 27% and 28% of patients in old and new eras respectively. STR without XRT was performed in 20% and 29% of patients in old and new era respectively. Cyst drainage and adjuvant radiotherapy were performed in 49% of patients in the old era while no patients in the new era underwent such conservative management. Aggressive surgical resection was associated with a higher risk of worsening visual outcomes (20% vs 16%), panhypopituitarism and diabetes insipidus (86% vs 53%), psycho-social impairment (42% vs 26%), and new-onset obesity (33% vs 22%). The mortality rate was higher in the old era in comparison with that of the new one (9% vs 2%). Conclusion There was a paradigm shift in management strategies of pediatric craniopharyngioma over the past six decades which in turn affected the long-term outcomes and quality of life of patients.