ESPE2021 Free Communications Neuroendocrinology (6 abstracts)
A novel clinical risk score that can accurately predict recurrence of craniopharyngioma - a multicentre cohort study
Nikolina Kyprianou 1 , James Blackburn 1 , Rachael Tan 1 , Gaetano Bulfamante 2 , Valentina Massa 2 , Federico Roncaroli 3 , Teresa Ribalta 4 , Jane Evanson 5 , Marta Korbonits 1 , Mehul Dattani 6 , Ashutosh Rai 7 , Prakamya Gupta 7 , Pinaki Dutta 7 , Anil Bhansali 7 , Pravin Salunke 7 , Danda Pani 7 , Tanja Skoric 8 , Darko Kastelan 8 , Kanna Gnanalingham 9 , Rod Mitchell 10 , Antonio Bulfamante 2 , Jesus Argente 11 , Nicolas Goycoolea 12 , Jorge Torales 13 , Betina Biagetti 14 , Laura Audi 15 , Eugenia Resmini 16 , Susan Webb 16 , Ritika Kapoor 17 , Christopher Chandler 18 , Bassel Zebian 18 , Nick Thomas 18 , Nicolas Sampron 19 , Dimitrios Paraskevopoulos 20 , Cristina Preda 21 , Amar Ahmad 22 , Evelien F Pease Gevers 23 & Carles Gaston-Massuet 1
1Centre for Endocrinology, William Harvey Research Institute, Barts & the London School of Medicine and Dentistry, Queen Mary University of London, London, United Kingdom.;2Department of Health Science, Universita deglu Studi de Milano, Milan, Italy.;3Cellular Pathology, Salford Royal Foundation Trust, Salford, United Kingdom.;4Hospital de San Joan de Deu, Barcelona, Spain.;5Neuroradiology Department, Barts Health NHS Trust, London, United Kingdom.;6Genetics and Genomic Medicine Department, Institute of Child Health, Great Ormond Street Hospital, London, United Kingdom.;7Nehru Hospital, Chandigarh, India.;8University Hospital of Zagreb, Zagreb, Croatia.;9Salford Royal NHS foundation Trust, Salford, United Kingdom.;10Royal Infirmary of Edinburg, Edinburgh, United Kingdom.;11Hospital del Nino Jesus, Madrid, Spain.;12Instituto de Neurocirugia Asenjo, Santiago, Chile.;13Neurosurgery Department, Hospital Clinic of Barcelona, Barcelona, Spain.;14Endocrinology Department Vall d’Hebron University Hospital, Barcelona, Spain.;15Growth and Development Research Unit, Vall d’Hebron Research Institute (VHIR), Center for Biomedical Research on Rare Diseases (CIBERER), Barcelona, Spain.;16Instituto de Salud Carlos III, Barcelona, Spain. Department of Medicine/Endocrinology, IIB-SPau, Hospital Sant Pau, UAB, Ciberer U747, Barcelona, Spain.;17Paediatric Endocrinology, Kings College Hospital NHS Trust, London, United Kingdom.;18Neurosurgery, Kings College Hospital NHS Trust, London, United Kingdom.;19Servicio de Neurocirugía Hospital Universitario Donostia, San Sebastian, United Kingdom.;20Department of Neurosurgery, Barts Health NHS Trust, London, United Kingdom.;21University Hospital St Spiridon, Iasi, Romania.;22Cancer Intelligence Department, Cancer Research UK, London, United Kingdom.;23Department of Paediatric Endocrinology, Royal London Children’s Hospital London, Barts Health NHS Trust, London, United Kingdom
Background: Recurrence of craniopharyngiomas influences mortality. Apart from the extent of surgical resection, few clinical parameters have been consistently shown to be associated with recurrence. Radical resection is difficult due to infiltration of surrounding tissue and unacceptable morbidity. Predictors of recurrence are therefore needed.
Aim: To establish a multinational cohort of patients with craniopharyngioma and employ their clinical parameters to design a clinical tool that can predict the risk of recurrence.
Methods: 225 patients from 15 centres (8 countries) participated in our prospective and retrospective observational cohort study. Tumour subtyping was performed by three histopathologists. Detailed clinical and neuroimaging data was collected. Statistical analyses were performed using R software; the primary end-point for prediction was ‘time-to-first-recurrence’.
Results: 47% were <18 years; 89% were adamantinomatous. Median age in children was 10.4 (IQR 6.3-13.2) and adults 46.1 (IQR 31.7-58.5). 56% had a recurrence with median ‘time-to-first-recurrence’ of 23 months (IQR 9–44). A multivariate ridge Cox proportional hazards regression model was performed using age, gender and clinical parameters as risk predictors of time-to-first-recurrence (diagnosis decade; symptom duration; tumour subtype, size, consistency, location; hypothalamic invasion; endocrinopathies; transsphenoidal/craniotomy; complete/incomplete resection; radiotherapy). A risk-score was computed as the linear predictor of the fitted multivariate Cox model. For each year post-treatment and for up to 10 years, a risk score cut-off value with a sensitivity nearest to 90% was selected, which had an accompanying specificity. At 5-year follow-up, a cut-off value of 0.463 for the score gives a sensitivity of 89.9% (95% CI: 84-95%) and a specificity of 50.4% (95% CI: 39-64%). The corresponding area under the curve (AUC) at 5-year follow-up is 0.776 (95% CI: 0.701-0.867). The AUC is maintained above 80% for up to 10-year follow-up indicating a good prognostic ability of the score. A univariate Cox regression model showed that for every increase in the risk score by 1 unit, the IQR Hazard Ratio is 2.052 (95% CI: 1.663-2.532, LR χ2 49.742; P = 1.8e-12) with a Harrell’s C-index of 0.659 (95%CI: 0.605-0.712) further indicating good predictive performance. Kaplan–Meier curves showed that radiotherapy resulted in significantly longer recurrence-free-survival (P = 0.00046).
Conclusion: This study is the first to use a large comprehensive clinical dataset that combines several clinical features to design a model that predicts the risk of craniopharyngioma recurrence. This model facilitates the identification of risk factors and following external validation, could be used in clinical practice to aid more individualised management.
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