RESUMEN
Childhood cancer survivors (CCS) experience higher hospitalization rates compared to the general population for neoplasms, circulatory diseases, endocrine/nutritional/metabolic diseases and eye disorders. We studied trends in hospitalization rates and associated patient and treatment-specific risk factors for diagnosis subgroups among these four diseases. We performed medical record linkage of a ≥5-year CCS cohort with national registers, and obtained a random reference sample matched on age, gender and calendar year per CCS. For each diagnosis subgroup we compared hospitalization rates and trends over time in CCS and the reference population. Further, we analyzed risk factors for hospitalizations within the four CCS diagnosis groups. We used multivariate Poisson regression for all models. We retrieved hospitalization data from 1382 CCS and 26,583 reference persons. CCS had increased hospitalization rates for almost all diagnosis subgroups examined. Hospitalization rates for endocrine/nutritional/metabolic diseases appeared to increase with longer time since primary cancer diagnosis up to 30 years after primary cancer diagnosis. Survivors initially treated with radiotherapy had increased hospitalization rates for neoplasms (P < 0.001), those initially treated with anthracyclines (2.5 [1.1-5.5]) and radiotherapy to thorax and/or abdomen (9.3 [2.4-36.6]) had increased hospitalization rates for diseases of the circulatory system, and those initially treated with radiotherapy to head and/or neck had increased hospitalization rates for endocrine/nutritional/metabolic diseases (6.7 [3.5-12.7]) and diseases of the eye (3.6 [1.5-8.9]). Our study highlights that long-term health problems resulting in hospitalizations are still clinically relevant later in life of CCS. The identified treatment-related risk factors associated with hospitalizations support targeted follow-up care for these risk groups of CCS.
Asunto(s)
Adultos Sobrevivientes de Eventos Adversos Infantiles/psicología , Supervivientes de Cáncer/psicología , Hospitalización/tendencias , Adulto , Antraciclinas/efectos adversos , Femenino , Humanos , Masculino , Registro Médico Coordinado , Persona de Mediana Edad , Radioterapia/efectos adversos , Factores de Riesgo , Adulto JovenRESUMEN
Hospitalization rates over time of childhood cancer survivors (CCS) provide insight into the burden of unfavorable health conditions on CCS and health care resources. The objective of our study was to examine trends in hospitalizations of CCS and risk factors in comparison with the general population. We performed a medical record linkage study of a cohort of 1564 ≥five-year CCS with national registers. We obtained a random sample of the general population matched on year of birth, gender and calendar year per CCS retrieved. We quantified and compared hospitalization rates of CCS and reference persons from 1995 until 2005, and we analyzed risk factors for hospitalization within the CCS cohort with multivariable Poisson models. We retrieved hospitalization information from 1382 CCS and 25583 reference persons. The overall relative hospitalization rate (RHR) was 2.2 (95%CI:1.9-2.5) for CCS compared to reference persons. CCS with central nervous system and solid tumors had highest RHRs. Hospitalization rates in CCS were increased compared to reference persons up to at least 30 years after primary diagnosis, with highest rates 5-10 and 20-30 years after primary cancer. RHRs were highest for hospitalizations due to neoplasms (10.7; 95%CI:7.1-16.3) and endocrine/nutritional/metabolic disorders (7.3; 95%CI:4.6-11.7). Female gender (P<0.001), radiotherapy to head and/or neck (P<0.001) or thorax and/or abdomen (P = 0.03) and surgery (P = 0.01) were associated with higher hospitalization rates in CCS. In conclusion, CCS have increased hospitalization rates compared to the general population, up to at least 30 years after primary cancer treatment. These findings imply a high and long-term burden of unfavorable health conditions after childhood cancer on survivors and health care resources.
Asunto(s)
Costo de Enfermedad , Hospitalización/estadística & datos numéricos , Modelos Estadísticos , Neoplasias/rehabilitación , Sistema de Registros , Sobrevivientes/estadística & datos numéricos , Adulto , Estudios de Casos y Controles , Femenino , Cabeza/efectos de la radiación , Humanos , Estudios Longitudinales , Masculino , Registro Médico Coordinado , Persona de Mediana Edad , Cuello/efectos de la radiación , Neoplasias/clasificación , Neoplasias/patología , Neoplasias/terapia , Países Bajos , Factores de Riesgo , Factores Sexuales , Tórax/efectos de la radiaciónRESUMEN
In the Netherlands, the postal code is needed to study hospitalizations of individuals in the nationwide hospitalization register. Studying hospitalizations longitudinally becomes troublesome if individuals change address. We aimed to report on the feasibility and validity of a two-step medical record linkage approach to examine longitudinal trends in hospitalizations and mortality in a study cohort. First, we linked a study cohort of 1564 survivors of childhood cancer with the Municipal Personal Records Database (GBA) which has postal code history and mortality data available. Within GBA, we sampled a reference population matched on year of birth, gender and calendar year. Second, we extracted hospitalizations from the Hospital Discharge Register (LMR) with a date of discharge during unique follow-up (based on date of birth, gender and postal code in GBA). We calculated the agreement of death and being hospitalized in survivors according to the registers and to available cohort data. We retrieved 1477 (94%) survivors from GBA. Median percentages of unique/potential follow-up were 87% (survivors) and 83% (reference persons). Characteristics of survivors and reference persons contributing to unique follow-up were comparable. Agreement of hospitalization during unique follow-up was 94% and agreement of death was 98%. In absence of unique identifiers in the Dutch hospitalization register, it is feasible and valid to study hospitalizations and mortality of individuals longitudinally using a two-step medical record linkage approach. Cohort studies in the Netherlands have the opportunity to study mortality and hospitalization rates over time. These outcomes provide insight into the burden of clinical events and healthcare use in studies on patients at risk of long-term morbidities.
Asunto(s)
Bases de Datos Factuales , Hospitalización , Neoplasias/mortalidad , Sistema de Registros , Adolescente , Niño , Preescolar , Supervivencia sin Enfermedad , Femenino , Humanos , Lactante , Recién Nacido , Masculino , Neoplasias/terapia , Países Bajos/epidemiología , Estudios Retrospectivos , Tasa de SupervivenciaRESUMEN
PURPOSE: To evaluate the prevalence and severity of clinical adverse events (AEs) and treatment-related risk factors in childhood cancer survivors treated with cranial radiation therapy (CRT), with the aim of assessing dose-effect relationships. METHODS AND MATERIALS: The retrospective study cohort consisted of 1362 Dutch childhood cancer survivors, of whom 285 were treated with CRT delivered as brain irradiation (BI), as part of craniospinal irradiation (CSI), and as total body irradiation (TBI). Individual CRT doses were converted into the equivalent dose in 2-Gy fractions (EQD(2)). Survivors had received their diagnoses between 1966 and 1996 and survived at least 5 years after diagnosis. A complete inventory of Common Terminology Criteria for Adverse Events grade 3.0 AEs was available from our hospital-based late-effect follow-up program. We used multivariable logistic and Cox regression analyses to examine the EQD(2) in relation to the prevalence and severity of AEs, correcting for sex, age at diagnosis, follow-up time, and the treatment-related risk factors surgery and chemotherapy. RESULTS: There was a high prevalence of AEs in the CRT group; over 80% of survivors had more than 1 AE, and almost half had at least 5 AEs, both representing significant increases in number of AEs compared with survivors not treated with CRT. Additionally, the proportion of severe, life-threatening, or disabling AEs was significantly higher in the CRT group. The most frequent AEs were alopecia and cognitive, endocrine, metabolic, and neurologic events. Using the EQD(2), we found significant dose-effect relationships for these and other AEs. CONCLUSION: Our results confirm that CRT increases the prevalence and severity of AEs in childhood cancer survivors. Furthermore, analyzing dose-effect relationships with the cumulative EQD(2) instead of total physical dose connects the knowledge from radiation therapy and radiobiology with the clinical experience.
Asunto(s)
Irradiación Craneana/efectos adversos , Sobrevivientes , Adolescente , Adulto , Niño , Preescolar , Fraccionamiento de la Dosis de Radiación , Relación Dosis-Respuesta en la Radiación , Femenino , Neoplasias de Cabeza y Cuello/epidemiología , Neoplasias de Cabeza y Cuello/etiología , Humanos , Lactante , Recién Nacido , Masculino , Neoplasias Inducidas por Radiación/epidemiología , Neoplasias Primarias Secundarias/epidemiología , Neoplasias Primarias Secundarias/etiología , Prevalencia , Traumatismos por Radiación/epidemiología , Análisis de Regresión , Estudios Retrospectivos , Factores de Riesgo , Índice de Severidad de la Enfermedad , Irradiación Corporal Total/efectos adversos , Adulto JovenRESUMEN
BACKGROUND: Modern WT management consist of ample chemotherapy, nephron-sparing surgery, and, when indicated, radiotherapy. Survivors may develop renal failure or secondary tumors due to anticancer treatment. We analyzed long-term outcome (follow-up >5 years) after bilateral Wilms tumor (BWT) treatment with respect to survival, renal function, and secondary malignancies. METHODS: From 41 patients (23 females, 28 synchronous tumors) diagnosed with BWT between 1967 and 2007, 25 (18 females, 14 synchronous) with a follow-up >5 years could be included. Of this subgroup, median age at diagnosis was 1.64 years (range 0.27-5.35), and at maximum follow-up 14.99 years (range 5.40-33.99). Data were retrospectively collected and analyzed. RESULTS: One patient (4%) died 17.75 years after diagnosis, five (20%) had renal transplants: 3/5 after bilateral nephrectomy for Denys-Drash syndrome (DDS), and 2/5 for ESRD after an interval of 7 and 18 years, respectively. All transplanted patients remained in CR. Another three patients developed mild renal insufficiency (creatinine levels 1.3, 1.8, and 2.8 mg/100 ml, respectively; N = 0.5-1.2), combined with hypertension in 1; neither of them was transplanted. Sixteen (64%) had normal renal function and were in CR. Long-term renal function appeared significantly better after bilateral nephron sparing surgery (NSS) then after other surgical procedures (P < 0.0001). Seven secondary tumors were found in five (20%) patients, one of whom had a DDS. CONCLUSION: Long-term 10-year overall survival was 78%. There was significant morbidity (13/25, 52%), in terms of renal failure (8/25, 32%) including renal transplantation (5/25, 20%), and secondary tumors (5/25). These findings necessitate long-term follow-up beyond childhood. Future work should be directed at reducing the harmful effects of treatment, including the increased use of NSS.
Asunto(s)
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Neoplasias Renales/terapia , Neoplasias Primarias Múltiples/terapia , Tumor de Wilms/terapia , Adolescente , Adulto , Niño , Terapia Combinada , Femenino , Estudios de Seguimiento , Humanos , Pruebas de Función Renal , Masculino , Nefrectomía , Dosificación Radioterapéutica , Estudios Retrospectivos , Tasa de Supervivencia , Factores de Tiempo , Resultado del Tratamiento , Adulto JovenRESUMEN
PURPOSE: To evaluate the prevalence and severity of adverse events (AEs) and treatment-related risk factors in long-term Wilms' tumor (WT) survivors, with special attention to radiotherapy. METHODS AND MATERIALS: The single-center study cohort consisted of 185 WT survivors treated between 1966 and 1996, who survived at least 5 years after diagnosis. All survivors were invited to a late-effects clinic for medical assessment of AEs. AEs were graded for severity in a standardized manner. Detailed radiotherapy data enabled us to calculate the equivalent dose in 2 Gy fractions (EQD(2)) to compare radiation doses in a uniform way. Risk factors were evaluated with multivariate logistic regression analysis. RESULTS: Medical follow-up was complete for 98% of survivors (median follow-up, 18.9 years; median attained age, 22.9 years); 123 survivors had 462 AEs, of which 392 had Grade 1 or 2 events. Radiotherapy to flank/abdomen increased the risk of any AE (OR, 1.08 Gy(-1) [CI, 1.04-1.13]). Furthermore, radiotherapy to flank/abdomen was associated with orthopedic events (OR, 1.09 Gy(-1) [CI, 1.05-1.13]) and second tumors (OR, 1.11 Gy(-1) [CI, 1.03-1.19]). Chest irradiation increased the risk of pulmonary events (OR, 1.14 Gy(-1) [CI, 1.06-1.21]). Both flank/abdominal and chest irradiation were associated with cardiovascular events (OR, 1.05 Gy(-1) [CI, 1.00-1.10], OR, 1.06 Gy(-1) [CI, 1.01-1.12]) and tissue hypoplasia (OR, 1.17 Gy(-1) [CI, 1.10-1.24], OR 1.10 Gy(-1) [CI, 1.03-1.18]). CONCLUSION: The majority of AEs, overall as well as in irradiated survivors, were mild to moderate. Nevertheless, the large amount of AEs emphasizes the importance of follow-up programs for WT survivors.
Asunto(s)
Antraciclinas/efectos adversos , Neoplasias Renales/radioterapia , Traumatismos por Radiación/epidemiología , Sobrevivientes , Tumor de Wilms/terapia , Adolescente , Adulto , Huesos/efectos de la radiación , Sistema Cardiovascular/efectos de los fármacos , Sistema Cardiovascular/efectos de la radiación , Niño , Preescolar , Factores Epidemiológicos , Fertilidad/efectos de la radiación , Humanos , Lactante , Riñón/efectos de la radiación , Neoplasias Renales/mortalidad , Pulmón/efectos de la radiación , Neoplasias Inducidas por Radiación/epidemiología , Neoplasias Primarias Secundarias/epidemiología , Traumatismos por Radiación/clasificación , Traumatismos por Radiación/mortalidad , Dosificación Radioterapéutica , Índice de Severidad de la Enfermedad , Tumor de Wilms/mortalidad , Adulto JovenRESUMEN
The information centre of the Emma Children's Hospital AMC (EKZ AMC) is a specialised information centre where paediatric patients and persons involved with the patient can ask questions about all aspects of disease and its social implications. The aim of the study was to evaluate the question-answer service of this information centre in order to determine the role of a specialised information centre in an academic children's hospital, identify the appropriate resources for the service and potential positive effects. For this purpose, a case management system was developed in MS ACCESS. The characteristics of the requester and the question, the time it took to answer questions, the information sources used and the extent to which we were able to answer the questions were registered. The costs of the service were determined. We analysed all questions that were asked in the year 2007. Fourteen hundred thirty-four questions were asked. Most questions were asked by parents (23.3%), healthcare workers (other than nurses; 16.5%) and nurses (15.3%). The scope of the most frequently asked questions include disease (20.2%) and treatment (13.0%). Information on paper was the main information source used. Most questions could be solved within 15 min. Twelve percent to 28% of total working hours are used for the question-answer service. Total costs including staff salary are rather large. In conclusions, taking over the task of providing additional medical information and by providing readily available, good quality information that healthcare professionals can use to inform their patients will lead to less time investment of these more expensive staff members. A specialised information service can anticipate on the information need of parents and persons involved with the paediatric patient. It improves information by providing with relatively simple resources that has the potential to improve patient and parent satisfaction, coping and medical results. A specialised information centre is therefore a valuable and affordable asset to an academic children's hospital.
Asunto(s)
Información de Salud al Consumidor , Servicios de Información , Pediatría , Niño , Información de Salud al Consumidor/economía , Costos y Análisis de Costo , Hospitales Pediátricos , Humanos , Servicios de Información/economía , Evaluación de Necesidades , Países Bajos , Evaluación de Programas y Proyectos de SaludRESUMEN
BACKGROUND: Clinical data and data on outcome of extra-osseous Ewing tumors are scarce. PROCEDURE: After a search for Ewing tumors in the database of a single institution over a period of 20 years, 16 out of 192 cases were found to have extra-osseous primary tumors. RESULTS: Ages at initial diagnosis ranged from 2.5 to 17 years. Follow-up period ranged from 4 months to 24.8 years (mean 8.4 years). Eleven patients were treated according to protocols for Ewing tumors, while in 4 cases soft tissue protocols were used. In a single patient only surgery was done. Two patients had progressive disease despite chemotherapy; a third patient had only tumor response on the initial 2 chemotherapy courses. All 3 patients with initially metastatic disease died. One patient developed a second malignancy. Overall survival at 5 years was 75%. Event-free survival (EFS) at 5 years was 68%; for nonmetastatic patients 5-year EFS was 83%. CONCLUSION: The authors conclude that nonmetastasized extra-osseous Ewing tumors have a prognosis at least similar to that of osseous Ewing tumors.
Asunto(s)
Neoplasias Óseas/tratamiento farmacológico , Neoplasias Óseas/mortalidad , Sarcoma de Ewing/tratamiento farmacológico , Sarcoma de Ewing/mortalidad , Adolescente , Antineoplásicos Alquilantes/uso terapéutico , Antineoplásicos Fitogénicos/uso terapéutico , Niño , Preescolar , Dactinomicina/uso terapéutico , Femenino , Estudios de Seguimiento , Humanos , Ifosfamida/uso terapéutico , Estimación de Kaplan-Meier , Masculino , Pronóstico , Inhibidores de la Síntesis de la Proteína/uso terapéutico , Resultado del Tratamiento , Vincristina/uso terapéuticoRESUMEN
BACKGROUND: Ewing sarcomas (ES) of the chest wall are rare. Local recurrences occur in approximately 20% of these patients; however literature on this topic is scarce. Our aim was to analyze the influence of the extent of surgical resection on outcome, and to find positive prognostic factors for survival. PROCEDURE: A retrospective analysis of all patients who were under 18 and treated between 1977 and 2006 for Ewing sarcoma of the chest wall was performed. RESULTS: Twenty-seven patients with ES were diagnosed at our institution; 14 males and 13 females. Twenty-three patients underwent local resection, which was complete in 13. Nine patients had a wide resection, including more ribs than just the affected one. Complete remission (CR) was achieved in 14/27 patients. Recurrence was observed in 13 patients: local recurrence (LR) and distant metastasis (DM) both in 7. One of the patients had both LR and DM. All patients with LR and two patients with DM only died of disease. Overall survival after 10 and 20 years was 73% and 47%, respectively. Complete resection, wide resection, gender, or age were no significant factors affecting outcome. CONCLUSIONS: Relapse carries a high mortality risk and none of the patients with LR eventually survived. Wide resection of at least a complete normal adjacent rib above and below the thoracic wall tumor as a fixed rule seems unnecessary.
Asunto(s)
Neoplasias Óseas/patología , Recurrencia Local de Neoplasia/patología , Sarcoma de Ewing/patología , Adolescente , Neoplasias Óseas/cirugía , Niño , Preescolar , Femenino , Humanos , Lactante , Masculino , Recurrencia Local de Neoplasia/mortalidad , Pronóstico , Estudios Retrospectivos , Sarcoma de Ewing/cirugía , Tasa de Supervivencia , Pared Torácica , Resultado del TratamientoRESUMEN
CONTEXT: Improved survival of children with cancer has been accompanied by multiple treatment-related complications. However, most studies in survivors of childhood cancer focused on only 1 late effect. OBJECTIVE: To assess the total burden of adverse health outcomes (clinical or subclinical disorders ["adverse events"]) following childhood cancer in a large cohort of childhood cancer survivors with long-term and complete medical follow-up. DESIGN, SETTING, AND POPULATION: Retrospective cohort study of 1362 five-year survivors of childhood cancer treated in a single institution in the Netherlands between 1966 and 1996. All survivors were invited to a late-effects clinic for medical assessment of adverse events. Adverse events occurring before January 2004 were graded for severity in a standardized manner. MAIN OUTCOME MEASURES: Treatment-specific prevalence of adverse events (according to severity) at end of follow-up and relative risk of high or severe burden of disease (> or =2 severe or > or =1 life-threatening or disabling adverse events) associated with various treatments. RESULTS: Medical follow-up was complete for 94.3% of survivors (median follow-up, 17.0 years). The median attained age at end of follow-up was 24.4 years. Almost 75% of survivors had 1 or more adverse events, and 24.6% had 5 or more adverse events. Furthermore, 40% of survivors had at least 1 severe or life-threatening or disabling adverse event. A high or severe burden of adverse events was observed in 55% of survivors who received radiotherapy only and 15% of survivors treated with chemotherapy only, compared with 25% of survivors who had surgery only (adjusted relative risks, 2.18 [95% confidence interval, 1.62-2.95] and 0.65 [95% confidence interval, 0.46-0.90], respectively). A high or severe burden of adverse events was most often observed in survivors of bone tumors (64%) and least often in survivors of leukemia or Wilms tumor (12% each). CONCLUSIONS: In young adulthood, a substantial proportion of childhood cancer survivors already has a high or severe burden of disease, particularly after radiotherapy. This underscores the need for lifelong risk-stratified medical surveillance of childhood cancer survivors.