Mark Wijnen1,2, Daniel S Olsson3,4, Marry M van den Heuvel-Eibrink2,5, Casper Hammarstrand3,4, Joseph A M J L Janssen1, Aart J van der Lely1, Gudmundur Johannsson3,4, Sebastian J C M M Neggers1,2. 1. Department of Medicine Section Endocrinology, Pituitary Centre Rotterdam, Erasmus University Medical Centre, Rotterdam, The Netherlands. 2. Department of Paediatric Oncology/Haematology Erasmus MC - Sophia Children's Hospital, Rotterdam, The Netherlands. 3. Department of Endocrinology Sahlgrenska University Hospital, Gothenburg, Sweden. 4. Department of Internal Medicine and Clinical Nutrition Institute of Medicine, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden. 5. Princess Maxima Centre for Paediatric Oncology Utrecht, The Netherlands.
Abstract
OBJECTIVE: Most studies in patients with craniopharyngioma did not investigate morbidity and mortality relative to the general population nor evaluated risk factors for excess morbidity and mortality. Therefore, the objective of this study was to examine excess morbidity and mortality, as well as their determinants in patients with craniopharyngioma. DESIGN: Hospital-based retrospective cohort study conducted between 1987 and 2014. METHODS: We included 144 Dutch and 80 Swedish patients with craniopharyngioma identified by a computer-based search in the medical records (105 females (47%), 112 patients with childhood-onset craniopharyngioma (50%), 3153 person-years of follow-up). Excess morbidity and mortality were analysed using standardized incidence and mortality ratios (SIRs and SMRs). Risk factors were evaluated univariably by comparing SIRs and SMRs between non-overlapping subgroups. RESULTS: Patients with craniopharyngioma experienced excess morbidity due to type 2 diabetes mellitus (T2DM) (SIR: 4.4, 95% confidence interval (CI): 2.8-6.8) and cerebral infarction (SIR: 4.9, 95% CI: 3.1-8.0) compared to the general population. Risks for malignant neoplasms, myocardial infarctions and fractures were not increased. Patients with craniopharyngioma also had excessive total mortality (SMR: 2.7, 95% CI: 2.0-3.8), and mortality due to circulatory (SMR: 2.3, 95% CI: 1.1-4.5) and respiratory (SMR: 6.0, 95% CI: 2.5-14.5) diseases. Female sex, childhood-onset craniopharyngioma, hydrocephalus and tumour recurrence were identified as risk factors for excess T2DM, cerebral infarction and total mortality. CONCLUSIONS: Patients with craniopharyngioma are at an increased risk for T2DM, cerebral infarction, total mortality and mortality due to circulatory and respiratory diseases. Female sex, childhood-onset craniopharyngioma, hydrocephalus and tumour recurrence are important risk factors.
OBJECTIVE: Most studies in patients with craniopharyngioma did not investigate morbidity and mortality relative to the general population nor evaluated risk factors for excess morbidity and mortality. Therefore, the objective of this study was to examine excess morbidity and mortality, as well as their determinants in patients with craniopharyngioma. DESIGN: Hospital-based retrospective cohort study conducted between 1987 and 2014. METHODS: We included 144 Dutch and 80 Swedish patients with craniopharyngioma identified by a computer-based search in the medical records (105 females (47%), 112 patients with childhood-onset craniopharyngioma (50%), 3153 person-years of follow-up). Excess morbidity and mortality were analysed using standardized incidence and mortality ratios (SIRs and SMRs). Risk factors were evaluated univariably by comparing SIRs and SMRs between non-overlapping subgroups. RESULTS:Patients with craniopharyngioma experienced excess morbidity due to type 2 diabetes mellitus (T2DM) (SIR: 4.4, 95% confidence interval (CI): 2.8-6.8) and cerebral infarction (SIR: 4.9, 95% CI: 3.1-8.0) compared to the general population. Risks for malignant neoplasms, myocardial infarctions and fractures were not increased. Patients with craniopharyngioma also had excessive total mortality (SMR: 2.7, 95% CI: 2.0-3.8), and mortality due to circulatory (SMR: 2.3, 95% CI: 1.1-4.5) and respiratory (SMR: 6.0, 95% CI: 2.5-14.5) diseases. Female sex, childhood-onset craniopharyngioma, hydrocephalus and tumour recurrence were identified as risk factors for excess T2DM, cerebral infarction and total mortality. CONCLUSIONS:Patients with craniopharyngioma are at an increased risk for T2DM, cerebral infarction, total mortality and mortality due to circulatory and respiratory diseases. Female sex, childhood-onset craniopharyngioma, hydrocephalus and tumour recurrence are important risk factors.
Authors: Hermann L Müller; Maithé Tauber; Elizabeth A Lawson; Jale Özyurt; Brigitte Bison; Juan-Pedro Martinez-Barbera; Stephanie Puget; Thomas E Merchant; Hanneke M van Santen Journal: Nat Rev Dis Primers Date: 2022-04-21 Impact factor: 52.329
Authors: Selveta S van Santen; Daniel S Olsson; Marry M van den Heuvel-Eibrink; Mark Wijnen; Casper Hammarstrand; Joseph A M J L Janssen; Gudmundur Johansson; Aart J van der Lely; Sebastian J C M M Neggers Journal: J Clin Endocrinol Metab Date: 2020-04-01 Impact factor: 5.958
Authors: Selveta S van Santen; Peter Wolf; Natalia Kremenevski; Cesar L Boguszewski; Hannes Beiglböck; Marta Fiocco; Mark Wijnen; Ville R Wallenius; Marry M van den Heuvel-Eibrink; Aart J van der Lely; Gudmundur Johannsson; Anton Luger; Michael Krebs; Michael Buchfelder; Patric J D Delhanty; Sebastian J C M M Neggers; Daniel S Olsson Journal: J Clin Endocrinol Metab Date: 2021-10-21 Impact factor: 5.958