Intracystic interferon-alpha in pediatric craniopharyngioma patients: an international multicenter assessment on behalf of SIOPE and ISPN (original) (raw)
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Interferon-alfa in the management of cystic craniopharyngioma in children under 5 years of age
Archives of Pediatric Neurosurgery, 2021
Introduction: the best therapeutic option for the management of craniopharyngioma in younger children remains controversial, ranging from complete surgical resection, partial surgical resection associated with radiotherapy and application of chemotherapeutic agents such as bleomycin and interferon-alfa. Objective: to verify the response to treatment with interferon-alfa via Ommaya reservoir in a group of children under 5 years of age with diagnosis of cystic craniopharyngioma. Methods: description of a case series through consecutive review of medical records of children under 5 years of age diagnosed with cystic adamantinomatous craniopharyngioma who had an Ommaya reservoir catheter surgically implanted for intratumoral application of interferon-?. Results: seven children aged 18 to 60 months (median 46 months) with the abovementioned diagnosis and treated with interferon-alfa between 2010 and 2019, according to a pre-established protocol, were identified. A reduction in tumor volu...
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.
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
Advances in the management of craniopharyngioma in children and adults
Radiology and Oncology
Background Childhood and adult-onset craniopharyngioma is a rare embryogenic tumor of the sellar, suprasellar, and parasellar region. Survival rates are high; however, tumor location and treatment sequalae including endocrine deficits, visual impairment, metabolic complications, cognitive and psychosocial deficits can significantly impair patient’s quality of life. There is considerable controversy regarding the optimal management of craniopharyngiomas. Subtotal resection of the tumor followed by targeted irradiation to avoid further hypothalamic damage is currently indicated. Novel insights in the tumor’s molecular pathology present the possibility for targeted therapy possibly decreasing the rate and severity of treatment-associated morbidity. Conclusions Craniopharyngioma should be seen as a chronic disease. To achieve optimal outcomes a multidisciplinary team of specialized neurosurgeons, neuro-radiologists, neuro-oncologists, pathologists and endocrinologists should be involved...
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...
Expert Review of Neurotherapeutics, 2018
Introduction: Childhood-onset craniopharyngiomas are rare embryonic tumors of lowgrade histological malignancy. Severe obesity, physical fatigue and psychosocial deficits due to hypothalamic tumor involvement have negative impact on quality of life. Initial pretreatment involvement of hypothalamic structures and/or treatment-related lesions result in sequelae clinically associated with impaired social and physical functionality and severe neuroendocrine deficiencies. Overall and progression-free survival rates are not associated with the degree of surgical resection. However, reduced overall survival rates were observed in patients with primary hypothalamic tumor involvement. Areas covered: This review discusses new perspectives on diagnostics, treatment and follow-up of patients with childhood-onset craniopharyngioma, which were mostly published after 2010 and presented at the 5th International Multidisciplinary Postgraduate Course on Childhood Craniopharyngioma, 19-22 April, 2018, at Bad Zwischenahn, Germany. Expert Commentary: Percutaneous radiooncological treatment options are effective in prevention of relapses and tumor progressions. Initial experience with proton beam therapy in childhood-onset craniopharyngioma patients shows promising results in terms of more protective radiological treatment. Recent reports on the molecular pathogenesis of craniopharyngioma open perspectives on the possibility of testing novel treatments targeting pathogenic pathways. As long as effective treatment options for hypothalamic syndrome are not available, hypothalamus-sparing treatment strategies are recommended.
Craniopharyngioma: a comparison of tumor control with various treatment strategies
Neurosurgical Focus, 2010
C raniopharyngiomas are the most common pituitary masses in children. They represent 6-13% of all childhood brain lesions and are classified as intracranial tumors of benign or unspecified behavior by pediatric cancer registries. 32,98 The Childhood Cancer Registry of Piedmont, Italy, has postulated an estimated incidence of 1.4 cases per million children per year with similar estimates provided by other cancer registries in Western countries. 32 Epidemiologically, craniopharyngiomas have a bimodal age distribution pattern with a peak between 5 and 14 years and in adults older than 65 years, although the tumor has been reported in all age groups. 125,274 Over the past few years Haupt et al. 98 have documented dramatic improvements in survival, but craniopharyngiomas continue to pose a challenge for Craniopharyngioma: a comparison of tumor control with various treatment strategies
Childhood Craniopharyngioma: A 22-Year Challenging Follow-Up in a Single Center
Hormone and metabolic research = Hormon- und Stoffwechselforschung = Hormones et metabolisme, 2018
Craniopharyngioma is a sellar/suprasellar benign tumor whose aggressiveness may imply in endocrine disturbances (hypothalamic obesity and hormone deficiencies). Fifty-seven patients were evaluated according to clinical characteristics, hypothalamic involvement, type of treatment, anthropometric variables, adiposity indexes (body mass index Z score category at diagnosis and post-treatment, total body fat, visceral adipose tissue, and metabolic syndrome components) and analyzed through multiple regression and logistic models. Patients were stratified according to growth hormone deficiency and recombinant human growth hormone use. Mean ages at diagnosis and at study evaluation were 9.6 and 16.6 years old, respectively. A set of 43/57 (75.4%) patients presented with important hypothalamic involvement, 24/57 (42.1%) received surgical treatment and cranial radiotherapy, and 8/57 (14%) interferon-α exclusively. Fifty-five patients (96.5%) were considered growth hormone deficient, and 26/57...