Daniel S Olsson1, Eva Andersson, Ing-Liss Bryngelsson, Anna G Nilsson, Gudmundur Johannsson. 1. Department of Endocrinology (D.S.O., A.G.N., G.J.), Sahlgrenska University Hospital, SE-413 45 Gothenburg, Sweden; Department of Occupational and Environmental Medicine (E.A.), Sahlgrenska University Hospital, SE-405 30 Gothenburg, Sweden; and Department of Occupational and Environmental Medicine (I.-L.B.), Örebro University Hospital, SE-701 85 Örebro, Sweden.
Abstract
CONTEXT: Craniopharyngiomas (CPs) in adults have been associated with excess mortality. OBJECTIVE: The aim of the study was to investigate mortality and morbidity in patients with childhood-onset and adult-onset CP. METHODS: PATIENTS with CP were identified and followed in Swedish national health registries, 1987 through 2011. The inclusion criteria for the CP diagnosis were internally validated against patient records in 28% of the study population. SETTINGS: This was a nationwide population-based study. PATIENTS: A total of 307 patients (151 men and 156 women) were identified and included (mean follow-up, 9 years; range, 0-25 years). The inclusion criteria had a positive predictive value of 97% and a sensitivity of 92%. INTERVENTION: There were no interventions. MAIN OUTCOME MEASURES: Standardized mortality ratios (SMRs) and standardized incidence ratios (SIRs) with 95% confidence intervals were calculated using the Swedish population as the reference. RESULTS: During the study, 54 patients died compared with the expected number of 14.1, resulting in an SMR of 3.2 (2.2-4.7) for men and 4.9 (3.2-7.2) for women. PATIENTS with childhood-onset (n = 106) and adult-onset (n = 201) CP had SMRs of 17 (6.3-37) and 3.5 (2.6-4.6), respectively. PATIENTS with hypopituitarism (n = 250), diabetes insipidus (n = 110), and neither of these (n = 54) had SMRs of 4.3 (3.1-5.8), 6.1 (3.5-9.7), and 2.7 (1.4-4.6), respectively. The SMR due to cerebrovascular diseases was 5.1 (1.7-12). SIRs were 5.6 (3.8-8.0) for type 2 diabetes mellitus, 7.1 (5.0-9.9) for cerebral infarction, 0.7 (0.2-1.7) for myocardial infarction, 2.1 (1.4-3.0) for fracture, and 5.9 (3.4-9.4) for severe infection. The SIR for all malignant tumors was 1.3 (0.8-2.1). CONCLUSIONS: This first nationwide population-based study of patients with CP demonstrated excess mortality that was especially marked in patients with childhood-onset disease and among women. Death due to cerebrovascular diseases was increased 5-fold. Hypopituitarism and diabetes insipidus were negative prognostic factors for mortality and morbidity. PATIENTS with CP had increased disease burden related to type 2 diabetes mellitus, cerebral infarction, fracture, and severe infection.
CONTEXT: Craniopharyngiomas (CPs) in adults have been associated with excess mortality. OBJECTIVE: The aim of the study was to investigate mortality and morbidity in patients with childhood-onset and adult-onset CP. METHODS:PATIENTS with CP were identified and followed in Swedish national health registries, 1987 through 2011. The inclusion criteria for the CP diagnosis were internally validated against patient records in 28% of the study population. SETTINGS: This was a nationwide population-based study. PATIENTS: A total of 307 patients (151 men and 156 women) were identified and included (mean follow-up, 9 years; range, 0-25 years). The inclusion criteria had a positive predictive value of 97% and a sensitivity of 92%. INTERVENTION: There were no interventions. MAIN OUTCOME MEASURES: Standardized mortality ratios (SMRs) and standardized incidence ratios (SIRs) with 95% confidence intervals were calculated using the Swedish population as the reference. RESULTS: During the study, 54 patients died compared with the expected number of 14.1, resulting in an SMR of 3.2 (2.2-4.7) for men and 4.9 (3.2-7.2) for women. PATIENTS with childhood-onset (n = 106) and adult-onset (n = 201) CP had SMRs of 17 (6.3-37) and 3.5 (2.6-4.6), respectively. PATIENTS with hypopituitarism (n = 250), diabetes insipidus (n = 110), and neither of these (n = 54) had SMRs of 4.3 (3.1-5.8), 6.1 (3.5-9.7), and 2.7 (1.4-4.6), respectively. The SMR due to cerebrovascular diseases was 5.1 (1.7-12). SIRs were 5.6 (3.8-8.0) for type 2 diabetes mellitus, 7.1 (5.0-9.9) for cerebral infarction, 0.7 (0.2-1.7) for myocardial infarction, 2.1 (1.4-3.0) for fracture, and 5.9 (3.4-9.4) for severe infection. The SIR for all malignant tumors was 1.3 (0.8-2.1). CONCLUSIONS: This first nationwide population-based study of patients with CP demonstrated excess mortality that was especially marked in patients with childhood-onset disease and among women. Death due to cerebrovascular diseases was increased 5-fold. Hypopituitarism and diabetes insipidus were negative prognostic factors for mortality and morbidity. PATIENTS with CP had increased disease burden related to type 2 diabetes mellitus, cerebral infarction, fracture, and severe infection.
Authors: Mohammed A Fouda; David Zurakowski; R Michael Scott; Karen J Marcus; Peter E Manley; Nicole J Ullrich; Laurie E Cohen; Liliana C Goumnerova Journal: Childs Nerv Syst Date: 2020-09-04 Impact factor: 1.475
Authors: M Wijnen; D S Olsson; M M van den Heuvel-Eibrink; V Wallenius; J A M J L Janssen; P J D Delhanty; A J van der Lely; G Johannsson; S J C M M Neggers Journal: Int J Obes (Lond) Date: 2016-10-31 Impact factor: 5.095
Authors: C Capatina; M Vintila; I Gherlan; A Dumitraşcu; A Caragheorgheopol; C Procopiuc; V Ciubotaru; C Poiana Journal: Acta Endocrinol (Buchar) Date: 2018 Oct-Dec Impact factor: 0.877