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1.
Br J Cancer ; 130(2): 260-268, 2024 02.
Artículo en Inglés | MEDLINE | ID: mdl-37993542

RESUMEN

BACKGROUND: Survivors of childhood cancer may face difficulties at school. We investigated whether childhood cancer affects attainment of upper secondary education, in a register-based cohort study from Denmark, Finland, and Sweden, where we limit bias from selection and participation. METHODS: From the national cancer registers, we identified all long-term survivors of childhood cancer diagnosed aged 0-14 years in 1971-2005 (n = 7629), compared them to matched population comparisons (n = 35,411) and siblings (n = 6114), using odds ratios (OR) and 95% confidence intervals (CI). RESULTS: Overall, 6127 survivors (80%) had attained upper secondary education by age 25, compared to 84% among comparison groups. Elevated OR for not attaining this level were mainly confined to survivors of central nervous system (CNS) tumours (ORSurv_PopComp2.05, 95%CI: 1.83-2.29). Other risk groups were survivors who had spent more time in hospital around cancer diagnosis and those who had hospital contacts in early adulthood, particularly psychiatric. Survivors of all cancer types were less likely to have attained upper secondary education without delay. CONCLUSIONS: Although survivors of childhood cancer experienced delays in their education, many had caught up by age 25. Except for survivors of CNS tumours, survivors attained upper secondary education to almost the same extent as their peers.


Asunto(s)
Supervivientes de Cáncer , Neoplasias del Sistema Nervioso Central , Neoplasias , Niño , Humanos , Adulto , Neoplasias/epidemiología , Estudios de Cohortes , Suecia/epidemiología , Finlandia/epidemiología , Escolaridad , Neoplasias del Sistema Nervioso Central/epidemiología , Sobrevivientes , Dinamarca/epidemiología
2.
Eur J Endocrinol ; 189(2): 190-198, 2023 Aug 02.
Artículo en Inglés | MEDLINE | ID: mdl-37542520

RESUMEN

OBJECTIVE: Previous studies have found that neurofibromatosis 1 (NF1) is associated with an increased risk for endocrine disorders, but no comprehensive overview of the risk for specific endocrine disorders has been published. We assessed endocrine morbidity in individuals with NF1 from information on hospital admissions, surgery for endocrine disorders, and relevant medication. DESIGN: A nationwide population registry-based cohort study. METHODS: We identified 2467 individuals with NF1 diagnosed between 1977 and 2013 from the Danish National Patient Register and the RAREDIS database and 20 132 randomly sampled age- and sex-matched population comparisons. Information on endocrine diseases was identified using registrations of discharge diagnoses, surgery, and medication prescriptions. The rates of endocrine disorders in individuals with NF1 were compared with those in the comparison cohort in Cox proportional hazard models. RESULTS: Individuals with NF1 had a higher rate than the comparison group of any endocrine discharge diagnosis (hazard ratio [HR] 1.72, 95% confidence interval [CI]: 1.58-1.87), endocrine-related surgery (2.03, 1.39-2.96), and prescribed medications (1.32, 1.23-1.42). Increased HRs were observed for diseases and surgical operations of several glands, including pheochromocytoma, and for osteoporosis, and osteoporotic fractures. Decreased rates were observed with drugs for type 2 diabetes. Women with NF1 had higher HRs for surgery of the ovaries, uterus, and sterilization, but lower rates of surgeries of cervix and prescriptions for birth control pills. CONCLUSIONS: Neurofibromatosis 1 is associated with a variety of endocrine disorders, surgery, and medication related to endocrine disease. Awareness of endocrine morbidity is important in the clinical follow-up of individuals with NF1.


Asunto(s)
Neoplasias de las Glándulas Suprarrenales , Diabetes Mellitus Tipo 2 , Enfermedades del Sistema Endocrino , Neurofibromatosis 1 , Humanos , Femenino , Neurofibromatosis 1/epidemiología , Estudios de Cohortes , Diabetes Mellitus Tipo 2/complicaciones , Morbilidad , Neoplasias de las Glándulas Suprarrenales/complicaciones , Enfermedades del Sistema Endocrino/epidemiología , Enfermedades del Sistema Endocrino/complicaciones
3.
Cancer Med ; 12(15): 16455-16468, 2023 08.
Artículo en Inglés | MEDLINE | ID: mdl-37305982

RESUMEN

BACKGROUND: Childhood cancer survivors face various adverse consequences. This Nordic register-based cohort study aimed to assess whether survivors of childhood cancer are more likely to have low income than their peers. METHODS: We identified 17,392 childhood cancer survivors diagnosed at ages 0 to 19 between 1971 and 2009 with 83,221 age-, sex-, and country-matched population comparisons. Annual disposable income at ages 20 to 50 years was retrieved from statistical offices (for 1990-2017) and categorized into low income and middle/high income. The number of transitions between income categories were assessed using binomial regression analyses. RESULTS: The prevalence of annual low income among childhood cancer survivors was 18.1% and 15.6% among population comparisons (risk ratio [RR] 1.17; 95% confidence interval [CI] 1.16-1.18). Compared to population comparisons, childhood cancer survivors were 10% (95% CI 8%-11%) less likely to transition from low to middle/high income and 12% (10%-15%) more likely to transition from middle/high to low income during follow-up. Among those initially in the low income category, survivors were 7% (95% CI 3%-11%) more likely to remain in the low income category. If the initial category was middle/high income, childhood cancer survivors were 10% (95% CI 8%-11%) less likely to remain in the middle/high income and 45% (37%-53%) more likely to transition to the low income category permanently. CONCLUSIONS: Childhood cancer survivors are at higher risk for low income in adulthood than their peers. These disparities might be reduced by continued career counseling along with support in managing within the social security system.


Asunto(s)
Supervivientes de Cáncer , Renta , Estatus Socioeconómico Bajo , Neoplasias , Estudios de Cohortes , Humanos , Masculino , Femenino , Adolescente , Adulto Joven , Adulto , Neoplasias/mortalidad , Recién Nacido , Lactante , Preescolar , Niño , Dinamarca , Finlandia , Suecia
4.
BMJ Open ; 12(9): e065340, 2022 09 20.
Artículo en Inglés | MEDLINE | ID: mdl-36127120

RESUMEN

PURPOSE: The Danish neurofibromatosis 1 (NF1) cohort was initiated to study health-related, socioeconomic and psychological consequences of living with the monogenetic disorder NF1 using a nationwide and population-based approach. PARTICIPANTS: The cohort includes all 2467 individuals in Denmark who were hospitalised with or due to NF1 from 1977 to 2013 or registered in the RAREDIS Database (1995-2013), a national clinical database for rare diseases, or both. A comparison cohort matched to individuals with NF1 on sex and date of birth was identified in the Civil Registration System (n=20 132). FINDINGS TO DATE: All cohort members were linked to the unique Danish registries to obtain information on hospital contacts, birth outcomes, education and partnership. A questionnaire was completed by 244 of the 629 adult cohort members with NF1 registered in the RAREDIS Database to evaluate the psychosocial and emotional burden. Further, neuropsychological tests were performed on 103 adult cohort members with NF1 and 38 adult population comparisons. To date, six studies have been published. Individuals with NF1 had an increased risk for (1) hospitalisation for disorders affecting all organ systems of the body throughout all decades of life, (2) psychiatric disorders, (3) attaining a short or medium long education and (4) not forming a life partner. Women with NF1 had an increased risk for spontaneous abortions and stillbirths. Finally, adults with NF1 had an impaired quality of life and a high need for professional support for physical, psychological and work-related problems, which was partly associated with disease severity and visibility. FUTURE PLANS: The cohort will regularly be updated with newly diagnosed patients in the RAREDIS Database as well as with outcome information in the Danish registries. New studies are in progress to assess other medical and socioeconomic dimensions of living with NF1.


Asunto(s)
Neurofibromatosis 1 , Adulto , Estudios de Cohortes , Dinamarca/epidemiología , Femenino , Humanos , Neurofibromatosis 1/epidemiología , Embarazo , Calidad de Vida , Sistema de Registros
5.
J Med Genet ; 59(3): 237-242, 2022 03.
Artículo en Inglés | MEDLINE | ID: mdl-33495305

RESUMEN

BACKGROUND: The probability of a pregnancy, live birth, stillbirth and abortion has never been assessed in women with neurofibromatosis 1 (NF1) in a large population-based study. METHODS: We included 1006 women (15-49 years) registered with NF1 in the Danish National Patient Registry or followed in two national Centers for Rare Diseases and 10 020 women from the Danish population. Information on pregnancy outcomes was ascertained from health registries. Cumulative incidence, mean cumulative count, hazard ratios (HRs) and proportion ratios (PRs) with 95% CIs were calculated. RESULTS: The cumulative incidence of a first pregnancy at age 50 years was slightly lower in women with NF1 (74%; 95% CI 70 to 77) than in population comparisons (78%; 95% CI 77 to 79). When all pregnancies were included, two pregnancies were expected per woman at age of 50 years, irrespective of a NF1 diagnosis. The hazard of a pregnancy did not differ between women with NF1 (HR 1.03; 95% CI 0.95 to 1.11) and the comparisons after adjustment for somatic and psychiatric disease. The proportion of pregnancies that resulted in a live birth was 63% (783/1252) among women NF1 and 68% (8432/12 465) among the comparisons, yielding a PR of 0.95 (95% CI 0.90 to 1.00). The proportions of stillbirths (PR 2.83; 95% CI 1.63 to 4.93) and spontaneous abortions (PR 1.40; 95% CI 1.09 to 1.79) were increased in women with NF1. CONCLUSIONS: A similar hazard for pregnancy was observed for women with NF1 and population comparisons after adjustment for potential medical consequences of NF1. However, women with NF1 experienced more spontaneous abortions and stillbirths.


Asunto(s)
Aborto Espontáneo , Neurofibromatosis 1 , Aborto Espontáneo/epidemiología , Estudios de Cohortes , Dinamarca/epidemiología , Femenino , Humanos , Masculino , Persona de Mediana Edad , Neurofibromatosis 1/complicaciones , Neurofibromatosis 1/epidemiología , Neurofibromatosis 1/genética , Embarazo , Resultado del Embarazo , Sistema de Registros , Mortinato/epidemiología
6.
Psychooncology ; 30(11): 1939-1947, 2021 11.
Artículo en Inglés | MEDLINE | ID: mdl-34260790

RESUMEN

OBJECTIVE: To compare the risk of depression after diagnostic workup for prostate cancer (PCa), regardless of the histopathologic outcome, with that of a cancer-free population. METHODS: A nationwide cohort of Danish men who had a prostatic biopsy sample in 1998-2011 was identified from the Danish Prostate Cancer Registry and compared to an age-matched cohort from the background population. Men with other cancers, major psychiatric disorder, or prior use of antidepressants were excluded. The risk of depression defined as hospital contact for depression or prescription for antidepressants was determined from cumulative incidence functions and multivariate Cox regression models. RESULTS: Of 54,766 men who underwent diagnostic workup for PCa, benign results were found for 21,418 and PCa was diagnosed in 33,347. During up to 18 years of follow-up, the adjusted hazard of depression was higher in men with PCa than in the background population, with the highest risk in the two years after diagnosis (hazard ratio (HR) 2.77, 95% CI 2.66-2.87). Comorbidity and lowest or highest income were significant risk factors for depression and the cumulative incidence was substantially higher in men with metastatic or high-risk disease. In men with benign histopathology the HR for depression was 1.22 (95% CI 1.14-1.31) in the first two years but no different from the background population after that. CONCLUSIONS: Diagnostic workup for PCa is associated with an increased risk of depression, mainly among men with a diagnosis of PCa. Clinicians should be aware of depressive symptoms in prostate cancer patients.


Asunto(s)
Depresión , Neoplasias de la Próstata , Estudios de Cohortes , Depresión/diagnóstico , Depresión/epidemiología , Depresión/etiología , Humanos , Masculino , Próstata/patología , Neoplasias de la Próstata/diagnóstico , Neoplasias de la Próstata/epidemiología , Neoplasias de la Próstata/patología , Sistema de Registros
7.
Am J Med Genet A ; 185(12): 3706-3716, 2021 12.
Artículo en Inglés | MEDLINE | ID: mdl-34327813

RESUMEN

The aim of this study was to assess the risks of psychiatric disorders in a large cohort of 905 individuals with NF1 and 7614 population comparisons matched on sex and year of birth. The cohort was linked to the Danish Psychiatric Central Research Register to ascertain information on hospital contacts for psychiatric disorders based on the International Classification of Diseases version 8 and 10. The hazard ratio (HR) for a first psychiatric hospital contact was higher in girls (4.19, 95% confidence interval [CI] 1.81-9.69) and boys with NF1 (5.02, 95% CI 3.27-7.69) <7 years of age than in the population comparisons. Both sexes had increased HRs for developmental disorders, including attention deficit/hyperactivity disorders, autism spectrum disorders, and intellectual disabilities in childhood. Females with NF1 had also increased HRs for unipolar depression, other emotional and behavioral disorders, and severe stress reaction and adjustment disorders in early adulthood. The HRs for psychoses, schizophrenia, bipolar disorders, and substance abuse were similar in individuals with NF1 and the population comparisons. Finally, the cumulative incidence of a first hospital contact due to any psychiatric disorder by age 30 years was 35% (95% CI 29-41) in females and 28% (95% CI 19-37) in males with NF1. Thus, screening for psychiatric disorders may be important for early diagnosis and facilitation of appropriate and effective treatment in individuals with NF1.


Asunto(s)
Trastornos Mentales/epidemiología , Neurofibromatosis 1/epidemiología , Trastornos Psicóticos/epidemiología , Trastorno por Déficit de Atención con Hiperactividad/complicaciones , Trastorno por Déficit de Atención con Hiperactividad/epidemiología , Trastorno por Déficit de Atención con Hiperactividad/fisiopatología , Trastorno del Espectro Autista/complicaciones , Trastorno del Espectro Autista/epidemiología , Trastorno del Espectro Autista/fisiopatología , Niño , Preescolar , Dinamarca/epidemiología , Trastorno Depresivo/complicaciones , Trastorno Depresivo/epidemiología , Trastorno Depresivo/fisiopatología , Femenino , Humanos , Lactante , Discapacidad Intelectual/complicaciones , Discapacidad Intelectual/epidemiología , Discapacidad Intelectual/fisiopatología , Clasificación Internacional de Enfermedades/normas , Masculino , Trastornos Mentales/complicaciones , Trastornos Mentales/fisiopatología , Neurofibromatosis 1/complicaciones , Neurofibromatosis 1/fisiopatología , Modelos de Riesgos Proporcionales , Trastornos Psicóticos/complicaciones , Trastornos Psicóticos/patología , Factores de Riesgo , Esquizofrenia/complicaciones , Esquizofrenia/epidemiología , Esquizofrenia/fisiopatología , Resultado del Tratamiento
8.
JNCI Cancer Spectr ; 5(3)2021 06.
Artículo en Inglés | MEDLINE | ID: mdl-34085001

RESUMEN

Background: Having a child diagnosed with cancer is a devastating experience that may affect parents' mental health. We aimed to assess the risk of hospital contacts for psychiatric disorders in parents of children with cancer. Methods: We conducted a nationwide population-based cohort study using Danish registry data. Parents of children diagnosed with cancer between 1982 and 2014 (n = 6689 mothers, n = 5509 fathers) were matched with comparison parents of cancer-free children (n = 67 544 mothers, n = 55 756 fathers). We used Cox proportional hazards models to estimate the risk of hospital contacts for any psychiatric disorder and specific disorders. Cox models were also used to investigate sociodemographic and cancer-related risk factors for psychiatric disorders. Results: Incidence rates of hospital contacts for any psychiatric disorder were 426 per 100 000 person-years in mothers of children with cancer and 345 per 100 000 person-years in comparison mothers. For fathers, the respective incidence rates were 260 and 262 cases per 100 000 person-years. Compared with parents of cancer-free children, mothers of children with cancer were at an increased risk of hospital contacts for any psychiatric disorder (hazard ratio = 1.23, 95% confidence interval = 1.12 to 1.36), whereas no elevated risk was seen in fathers (hazard ratio = 0.99, 95% confidence interval = 0.87 to 1.13). Among mothers, risks were particularly elevated for affective and stress-related disorders. Parents of deceased children and children diagnosed at a younger age were at particular risk of hospital contacts for psychiatric disorders. Conclusion: Hospital contacts for psychiatric disorders were overall rare. Health-care professionals should draw attention to subgroups of vulnerable parents to meet their needs of support and adequate treatment.


Asunto(s)
Hijo de Padres Discapacitados , Padre/psicología , Trastornos Mentales , Madres/psicología , Neoplasias/diagnóstico , Adolescente , Adulto , Factores de Edad , Niño , Preescolar , Estudios de Cohortes , Dinamarca/epidemiología , Padre/estadística & datos numéricos , Femenino , Hospitalización/estadística & datos numéricos , Humanos , Incidencia , Lactante , Masculino , Persona de Mediana Edad , Madres/estadística & datos numéricos , Neoplasias/psicología , Modelos de Riesgos Proporcionales , Sistema de Registros , Factores de Riesgo , Factores Socioeconómicos , Adulto Joven
9.
Eur Urol Oncol ; 4(4): 601-608, 2021 08.
Artículo en Inglés | MEDLINE | ID: mdl-31345731

RESUMEN

BACKGROUND: Development of depression in prostate cancer patients depends on multiple disease- and patient-related factors. OBJECTIVE: To investigate the risk of depression following radical prostatectomy focussing on the impact of surgery and subsequent treatment with salvage radiation or androgen deprivation therapy. DESIGN, SETTING, AND PARTICIPANTS: A population-based cohort study of 5570 men who underwent radical prostatectomy in Denmark from 1998 to 2011 was identified in the Danish Prostate Cancer Registry. OUTCOME MEASUREMENTS AND STATISTICAL ANALYSIS: Data on covariates and primary outcome defined as a hospital contact for depression or a redeemed antidepressant prescription were obtained from nationwide Danish registries. The risk of depression was evaluated using cumulative incidence functions and Cox models with time since surgery as an underlying time scale. Exposure to salvage procedures was included as time-varying covariates, and analyses were adjusted for confounders. RESULTS AND LIMITATIONS: The cumulative incidence of depression was increased in men who had undergone surgery compared with cancer-free men throughout follow-up of up to 18yr, particularly among men on androgen deprivation therapy. Compared with no subsequent treatment, the risk of depression was increased with subsequent androgen deprivation therapy (hazard ratio [HR] 1.8, 95% confidence interval [CI] 1.4-2.3), salvage radiation (HR 1.3, 95% CI 1.0-1.6), and the treatments combined (HR 2.2, 95% CI 1.8-2.8) after adjustments for age, year of surgery, income, and cohabitation status. Further adjustment for comorbidity hardly changed the estimates. CONCLUSIONS: Radical prostatectomy and subsequent salvage procedures increase the risk of depression, and men with subsequent androgen deprivation therapy are mainly at risk. Clinicians should thus be aware of depressive symptoms in patients receiving treatment for postsurgical relapse. PATIENT SUMMARY: In a population-based study, we found that radical prostatectomy and subsequent treatments with either radiation or endocrine manipulation significantly increased the risk of developing clinical depression.


Asunto(s)
Antagonistas de Andrógenos , Neoplasias de la Próstata , Antagonistas de Andrógenos/efectos adversos , Estudios de Cohortes , Depresión/epidemiología , Depresión/etiología , Humanos , Masculino , Recurrencia Local de Neoplasia , Prostatectomía/efectos adversos , Neoplasias de la Próstata/epidemiología , Neoplasias de la Próstata/cirugía , Sistema de Registros , Estudios Retrospectivos
10.
Am J Med Genet A ; 182(7): 1704-1715, 2020 07.
Artículo en Inglés | MEDLINE | ID: mdl-32484306

RESUMEN

Neurofibromatosis type 1 (NF1) is a genetic condition characterized by numerous somatic manifestations. The psychosocial burden in adults has rarely been studied. We examined the prevalence of self-reported impairment of quality of life (QoL), symptoms of anxiety and depression and need for support, associated with disease severity and visibility. We conducted a nationwide cross-sectional study of all 467 adults with NF1 diagnosed between 1977 and 2016 at one of the two national centers for rare diseases in Denmark. A total of 244 (56% response rate) completed a questionnaire that included standard measures of QoL, symptoms of depression and anxiety, indicators of disease-related severity, visibility, and need for professional support. Associations between disease severity and visibility and psychosocial burden were analyzed in descriptive and multivariate models. We observed impaired QoL (mean = 81.3; 95% CI, 76.2; 86.4); 19% reported symptoms of depression (mean = 5.7; SD = 5.4), and 15% reported anxiety (mean = 5.1; SD = 5.2) at a clinical level. Adults with NF1 also reported requiring professional support for physical, psychological, and work-related problems. Disease severity and (partly) visibility were significantly (p < .0001) associated with psychosocial well-being and a requirement for support. This study provides new understanding of the factors associated with impaired QoL, indicating that follow-up care should be optimized into adult life.


Asunto(s)
Ansiedad/etiología , Depresión/etiología , Neurofibromatosis 1/psicología , Calidad de Vida , Adolescente , Adulto , Anciano , Ansiedad/epidemiología , Ansiedad/psicología , Estudios Transversales , Dinamarca/epidemiología , Depresión/epidemiología , Depresión/psicología , Femenino , Humanos , Masculino , Persona de Mediana Edad , Neurofibromatosis 1/etiología , Prevalencia , Calidad de Vida/psicología , Adulto Joven
11.
Cancer ; 126(14): 3330-3340, 2020 07 15.
Artículo en Inglés | MEDLINE | ID: mdl-32449155

RESUMEN

BACKGROUND: Childhood cancer is a devastating experience for the family. The objective of the current study was to assess the impact of having a child with cancer on parental separation, divorce, and future family planning among families residing in Denmark. METHODS: The authors conducted a nationwide cohort study using Danish registry data. Parents of children diagnosed with cancer between 1982 and 2014 (7066 children and 12,418 case parents) were matched with 10 comparison parents of cancer-free children per case parent (69,993 children and 125,014 comparison parents). We used discrete-time Cox regression models to compare the risk of separation (end of cohabitation) and divorce between case and comparison parents, and to identify risk factors for separation and divorce among case parents only. Descriptive statistics were used to compare family planning between case and comparison parents. RESULTS: Case parents were found to have a slightly lower risk of separation (hazard ratio, 0.96; 95% confidence interval, 0.93-0.99) and divorce (hazard ratio, 0.92; 95% confidence interval, 0.87-0.97) than comparison parents. The authors found that case parents who were aged <45 years, with short education (an International Standard Classification of Education code indicating early childhood education, primary education, and lower secondary education), and who were unemployed were at an increased risk of separation and divorce. Moreover, the parents of children diagnosed with cancer at a young age (aged <15 years) were more likely to separate or divorce. No differences with regard to the total number of children and time to a next child after the cancer diagnosis were observed between case and comparison parents. CONCLUSIONS: Having a child with cancer was not associated with an overall adverse impact on parents' risk of separation or divorce and future family planning. These encouraging findings should be communicated to parents to support them along their child's cancer trajectory.


Asunto(s)
Salud Infantil , Divorcio , Servicios de Planificación Familiar , Neoplasias/epidemiología , Sistema de Registros , Adolescente , Adulto , Anciano , Estudios de Casos y Controles , Niño , Preescolar , Dinamarca/epidemiología , Escolaridad , Femenino , Estudios de Seguimiento , Humanos , Lactante , Masculino , Persona de Mediana Edad , Padres , Modelos de Riesgos Proporcionales , Factores de Riesgo , Clase Social , Desempleo , Adulto Joven
12.
Genet Med ; 22(6): 1069-1078, 2020 06.
Artículo en Inglés | MEDLINE | ID: mdl-32107470

RESUMEN

PURPOSE: The aim was to assess lifetime risk for hospitalization in individuals with neurofibromatosis 1 (NF1). METHODS: The 2467 individuals discharged with a diagnosis indicating NF1 or followed in a clinical center for NF1 were matched to 20,132 general population comparisons. Based on diagnoses in 12 main diagnostic groups and 146 subcategories, we calculated rate ratios (RRs), absolute excess risks (AERs), and hazard ratios for hospitalizations. RESULTS: The RR for any first hospitalization among individuals with NF1 was 2.3 (95% confidence interval 2.2-2.5). A high AER was seen for all 12 main diagnostic groups, dominated by disorders of the nervous system (14.5% of all AERs), benign (13.6%) and malignant neoplasms (13.4%), and disorders of the digestive (10.5%) and respiratory systems (10.3%). Neoplasms, nerve and peripheral ganglia disease, pneumonia, epilepsy, bone and joint disorders, and intestinal infections were major contributors to the excess disease burden caused by NF1. Individuals with NF1 had more hospitalizations and spent more days in hospital than the comparisons. The increased risk for any hospitalization was observed for both children and adults, with or without an associated cancer. CONCLUSION: NF1 causes an overall greater likelihood of hospitalization, with frequent and longer hospitalizations involving all organ systems throughout life.


Asunto(s)
Neurofibromatosis 1 , Adulto , Niño , Dinamarca/epidemiología , Hospitalización , Humanos , Longevidad , Neurofibromatosis 1/diagnóstico , Neurofibromatosis 1/epidemiología , Sistema de Registros
13.
Int J Cancer ; 147(4): 1006-1017, 2020 08 15.
Artículo en Inglés | MEDLINE | ID: mdl-31943199

RESUMEN

Having a child with cancer may affect the socioeconomic situation of the parents. We aimed to assess the impact of childhood cancer on parental working status and income and to identify determinants of adverse changes after the child's cancer diagnosis by calendar period. We conducted a nationwide cohort study using Danish registry data. Parents of children diagnosed with cancer in 1982-2014 (n = 12,418) were matched with comparison parents of cancer-free children (n = 125,014). We analysed annual working status (working/not working) and annual disposable income (lowest quintile/not lowest quintile) of case and comparison parents over a period of 10 years after diagnosis by calendar period (1982-1999 vs. 2000-2014). Logistic regression models were used to identify determinants of adverse changes after diagnosis. Mothers of children diagnosed in 1982-1999 were more likely not working or having a low income than comparison mothers up to 10 years after diagnosis. This risk of not working or low income was lower in mothers of children diagnosed in 2000-2014 compared to 1982-1999 in the first years after diagnosis (pinteraction < 0.05). We observed no consistent patterns among fathers. Low parental education, diagnosis of lymphoid leukaemia and younger age of the child at diagnosis were the main determinants of adverse changes in working status or income after diagnosis. Childhood cancer adversely interfered with parents' socioeconomic situation in the earlier calendar period, particularly among mothers. The absence of such an effect in more recent years emphasises the supportive role of a countries' welfare system alongside the general advances in childhood cancer treatment.


Asunto(s)
Empleo/estadística & datos numéricos , Renta/estadística & datos numéricos , Neoplasias/terapia , Padres , Sistema de Registros/estadística & datos numéricos , Factores Socioeconómicos , Adolescente , Niño , Preescolar , Estudios de Cohortes , Dinamarca , Empleo/economía , Femenino , Humanos , Lactante , Masculino , Neoplasias/diagnóstico , Adulto Joven
14.
J Clin Oncol ; 37(13): 1111-1119, 2019 05 01.
Artículo en Inglés | MEDLINE | ID: mdl-30860947

RESUMEN

PURPOSE: In this nationwide registry study, we investigated socioeconomic and structural patterns in referral to phase I cancer trials in a case-control study design. METHODS: Personal identification numbers on all Danish patients referred to the Danish Phase I Unit at Rigshospitalet from 2005 to 2016, and a control group matched on age, sex, type of cancer, year of diagnosis, and time from diagnosis to referral ensured individual-level linkage between several registries. We examined the association between nonclinical factors-indicators of socioeconomic position and distance to the Phase I Unit-and referral using a conditional logistic regression analysis adjusted for several clinical factors. Association between nonclinical factors and enrollment once referred was examined with a Cox proportional hazards regression analysis in an historical cohort study design. RESULTS: Complete data were available for 1,026 (84%) of 1,220 referred patients. Significantly decreased odds for referral were identified for patients with long distance to the Phase I Unit compared with short distance (adjusted odds ratio [OR], 0.35; 95% CI, 0.30 to 0.41), for less education (less than 9 years) compared with more (more than 12 years; OR, 0.69; 95% CI, 0.56 to 0.91), and for belonging to the lowest income quintile compared with the highest (OR, 0.78; 95% CI, 0.62 to 0.97). Medium education (9 to 12 years) compared with more, being outside the workforce compared with being within, and living alone compared with living with a partner were also negatively associated with referral. Among patients referred, 252 enrolled in a trial. Nonclinical factors were not associated with enrollment. CONCLUSION: On the basis of individual long-term registry data from an unselected cohort, novel anticancer therapies seem to be tested on a socially selected group of patients with cancer.


Asunto(s)
Neoplasias/epidemiología , Neoplasias/terapia , Derivación y Consulta/estadística & datos numéricos , Adulto , Anciano , Estudios de Casos y Controles , Dinamarca/epidemiología , Femenino , Humanos , Masculino , Persona de Mediana Edad , Neoplasias/economía , Selección de Paciente , Modelos de Riesgos Proporcionales , Derivación y Consulta/economía , Sistema de Registros , Factores Socioeconómicos
15.
Eur J Hum Genet ; 27(6): 857-868, 2019 06.
Artículo en Inglés | MEDLINE | ID: mdl-30820037

RESUMEN

Most research on psychosocial consequences of neurofibromatosis type 1 (NF1) has focused on the relationship between disease factors and cognitive functioning. NF1 may impair domains of learning and attention, resulting in low academic performance. This study is the first nationwide population-based cohort study to investigate educational attainment and delay in completing mandatory school by persons with NF1. Educational information was collected from 550 persons at the age of 30 (born 1965-1984). They were diagnosed with NF1 in Denmark and compared to a cohort of NF1-free persons matched on gender and age (n = 4295). Multinomial logistic models were applied to estimate odds ratios (ORs) for obtaining short (≤9 years) or medium (10-12 years) education compared to long education (>12 years) by the age of 30 years. We calculated the probability of graduating 9th year of mandatory school at different ages in 932 persons with NF1 and 7962 NF1-free persons (born 1965-2000) using quantile regression. The OR of educational completion for short- and medium-term education was three fold (95% CI 2.55-3.99) and 1.29 fold (95% CI 0.99-1.69) higher, respectively, for persons with NF1 than NF1-free persons after adjusting for birth year, gender, psychiatric and somatic morbidity and mother's education. Persons with NF1 were significantly delayed in graduating mandatory school education compared to NF1-free persons. When 90% of persons have graduated, persons with NF1 were 1.2 times older than the NF1-free persons. Experiencing delays in mandatory school likely affect further educational achievements and may impair employment and entering work force.


Asunto(s)
Cognición , Educación , Neurofibromatosis 1 , Sistema de Registros , Instituciones Académicas , Adolescente , Adulto , Niño , Estudios de Cohortes , Dinamarca , Femenino , Humanos , Masculino , Persona de Mediana Edad
16.
Eur J Hum Genet ; 27(6): 994-996, 2019 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-30923333

RESUMEN

Since the publication of the article, the authors noticed that 'NFI cohort' and 'NFI-free cohort' columns in the 'Autismg' and the 'Autism/ADHD' rows had been erroneously interchanged in Table 3. This has now been updated in the HTML and PDF of the original article.

17.
Neuro Oncol ; 21(1): 125-136, 2019 01 01.
Artículo en Inglés | MEDLINE | ID: mdl-29850875

RESUMEN

Background: A comprehensive overview of neurologic complications among survivors of central nervous system (CNS) tumors in childhood is lacking. We aimed to investigate the risk for these disorders in a large, population-based study with outcome measures from nationwide hospital registries. Methods: We identified 4858 five-year survivors with diagnoses of CNS tumor in childhood in Denmark, Iceland, Finland, and Sweden in 1943-2007, and 166658 matched population comparison subjects. Inpatient discharge diagnoses of neurologic disorders were used to calculate relative risks (RRs) and absolute excess risks (AERs). Results: A neurologic disorder was verified in 1309 survivors, while 92.4 were expected, yielding an overall RR of 14.2 (95% confidence interval [CI]: 13.3-15.1) and an AER of 20 hospitalizations per 1000 persons per year. The risks remained increased more than 20 years after diagnosis (RR: 6.3, 95% CI: 5.6-7.2; AER: 11, 9-12). The most frequent diagnoses were epilepsy (affecting 14.1% of all survivors) followed by hydrocephalus (9.5%) and paralytic syndromes (4.2%), with RRs of 28.7 (95% CI: 26.0-31.6), 243 (95% CI: 190-311), and 40.3 (95% CI: 33.1-49.2), respectively. Of these outcomes, 30%-40% were diagnosed prior to or synchronously with the CNS tumor. The survivors had highly increased RRs for infectious diseases of the CNS, disorders of cranial nerves, and degenerative diseases of the nervous system. Conclusions: Survivors of childhood CNS tumors are at markedly increased risk for neurologic disorders throughout their lives. Health care professionals must be aware of survivors who might benefit from preventive interventions and intensive follow-up.


Asunto(s)
Supervivientes de Cáncer/estadística & datos numéricos , Neoplasias del Sistema Nervioso Central/complicaciones , Hospitalización/estadística & datos numéricos , Enfermedades del Sistema Nervioso/epidemiología , Enfermedades del Sistema Nervioso/etiología , Sistema de Registros/estadística & datos numéricos , Adolescente , Adulto , Niño , Preescolar , Estudios de Cohortes , Femenino , Estudios de Seguimiento , Humanos , Incidencia , Lactante , Recién Nacido , Masculino , Persona de Mediana Edad , Enfermedades del Sistema Nervioso/patología , Pronóstico , Factores de Riesgo , Países Escandinavos y Nórdicos/epidemiología , Tasa de Supervivencia , Adulto Joven
18.
Int J Cancer ; 142(4): 671-680, 2018 02 15.
Artículo en Inglés | MEDLINE | ID: mdl-28971474

RESUMEN

Little is known about social inequalities in childhood cancer survival. We investigated the impact of family circumstances on survival from paediatric central nervous system (CNS) tumours in a nationwide, register-based cohort of Danish children. All children born between 1973 and 2006 and diagnosed with a CNS tumour before the age of 20 years (N = 1,261) were followed until 10 years from diagnosis. Using Cox proportional hazards models, the impact of various family characteristics on overall survival was estimated. Hazard ratios (HRs) for all CNS tumours combined did not show strong associations between survival and any family characteristic. Analyses by CNS tumour subtypes showed reduced survival for children with glioma when living outside of Copenhagen (HR 1.55; CI 1.03; 2.35). For embryonal CNS tumours, the number of full siblings was associated with worse survival (HR for having 3+ siblings 3.60; CI 1.52; 8.53) and a trend of better survival was observed for children with parents of younger age at child's diagnosis and poorer survival of children with parents of older age. Despite free and uniform access to health care services, some family circumstances appear to affect survival from specific CNS tumour types in Danish children. Further research is warranted to gain a more comprehensive understanding of the impact of family factors on childhood cancer survival in other populations and to elaborate underlying mechanisms and pathways of those survival inequalities observed.


Asunto(s)
Neoplasias del Sistema Nervioso Central/mortalidad , Composición Familiar , Sistema de Registros/estadística & datos numéricos , Adolescente , Adulto , Neoplasias del Sistema Nervioso Central/epidemiología , Niño , Preescolar , Estudios de Cohortes , Dinamarca/epidemiología , Femenino , Humanos , Lactante , Recién Nacido , Masculino , Pronóstico , Tasa de Supervivencia , Adulto Joven
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