Follow-up Of Childhood Cancer Survivors Research Articles

Protective effect of the dexrazoxane against cardiotoxicity of the anthracycline in the treatment of the childhood cancers.

e22001 Background: Cardiovascular diseases have become the most important cause of morbidity and mortality in the long-term follow-up of childhood cancer survivors. Dexrazoxane (DEX) is the only FDA-approved drug to prevent Anthracycline-induced cardiotoxicity. In this study, we aimed to evaluate the cardioprotective efficacy of DEX in patients using Anthracycline in the treatment of childhood cancer. Methods: 142 patients aged 0-18 years, who were treated with cancer diagnosis in Ankara University Faculty of Medicine, Department of Pediatrics, Department of Pediatric Oncology, and who had been treated with Anthracycline group agents, were evaluated retrospectively. The cardiac functions of 87 patients who received only Anthracycline between November 2009 and March 2019 and 55 patients who received DEX + Anthracycline between March 2019 and June 2021 were compared. An ejection fraction (EF) of less than 50% in cardiac evaluation was defined as 'cardiac dysfunction'. Results: The group that received anthracycline with DEX; While 50.9% were girls, 49.1% were boys, and the mean age was 98.42 months, 48.3% of the group that took only Anthracycline was girls and 51.7% were boys, with a mean age of 105.75 months. There was no difference between the two groups in terms of diagnosis distribution. EF values of both groups before anthracycline were normal. While none of the patients in the DEX + Anthracycline group developed cardiac dysfunction during the follow-up, 11 (12.6%) patients in the Anthracycline group with no DEX had cardiac problems. Six Of the 11 patients (54.5%) who developed cardiac dysfunction died(p=0.012). There was a significant difference between the two groups with or without DEX patients in the manner of cardiac dysfunction(p=0.006) and death(p=0.009). Cardiac dysfunction development and mortality rates were higher in patients using only Anthracycline. Conclusions: In this study, it has been shown that DEX is very effective for preventing Anthracycline-related cardiac dysfunction, which is one of the most important causes of mortality and morbidity in childhood cancer survivors.

The risk of valvular heart disease in the French Childhood Cancer Survivors’ Study: Contribution of dose-volume histogram parameters

Background and purposeValvular Heart Disease (VHD) is a known complication of childhood cancer after radiotherapy treatment. However, the dose-volume-effect relationships have not been fully explored. Materials and methodsWe obtained individual heart Dose Volume Histograms (DVH) for survivors of the French Childhood Cancer Survivors Study (FCCSS) who had received radiotherapy. We calculated the Mean Dose to the Heart (MHD) in Gy, as well as the heart DVH parameters (Vd Gy, which represents the percentage of heart volume receiving at least d Gy), fixing the thresholds to 0.1 Gy, 5 Gy, 20 Gy, and 40 Gy. We analyzed them furtherly in the subpopulation of the cohort that was treated with a dose lower than 5 Gy (V0.1Gy|V5Gy=0%), 20 Gy (V5Gy|V20Gy=0%), and 40 Gy (V20Gy|V40Gy=0%), respectively. We investigated their role in the occurrence of a VHD in this population-based observational cohort study using the Cox proportional hazard model, adjusting for age at cancer diagnosis and chemotherapy exposure. ResultsMedian follow-up was 30.6 years. Eighty-one patients out of the 7462 (1 %) with complete data experienced a severe VHD (grade ≥ 3). The risk of VHD increased along with the MHD, and it was associated with high doses to the heart (V40Gy < 50 %, hazard ratio (HR) = 7.96, 95 % CI: 4.26–14.88 and V20Gy|V40Gy=0% >50 %, HR = 5.03, 95 % CI: [2.35–10.76]). Doses 5–20 Gy to more than 50 % (V5Gy|V20Gy=0% >50 %) of the heart induced a marginally non-significant estimated risk. We also observed a remarkable risk increase with attained age. ConclusionsOur results provide new insight into the VHD risk that may impact current treatments and long-term follow-up of childhood cancer survivors.

Cardio-Oncology in Childhood: State of the Art.

Cardio-oncology is an increasingly important field of cardiology that focuses on the detection, monitoring, and treatment of cardiovascular disease (CVD) occurring during and after oncological treatments. The survival rate for childhood cancer patients has dramatically increased thanks to new treatment protocols and cardiovascular (CV) sequelae represent the third most frequent cause of mortality in surviving patients. This study aims to provide a complete and updated review of all the main aspects of cardio-oncology in childhood and to highlight the critical issues. The problem of CV complications in childhood cancer survivors raises the need to make an early diagnosis of cardiotoxicity by the new imaging and laboratory techniques in order to intervene promptly and to implement pharmacological strategies and lifestyle changes to reduce or even to prevent cardiac injury. Furthermore, a stratification of CV risk, also including new predisposing factors such as the presence of some genetic mutations, is of paramount importance before undertaking oncological treatments. Besides, a systematic and personalized planning of long-term follow-up is fundamental to ensure a transition from pediatric to adult hospital and to avoid missed or late diagnosis of cardiomyopathy. We reviewed the main risk factors for cardiotoxicity in children, both traditional and emerging ones: the mechanisms of toxicity of both old and new antineoplastic therapies, the techniques for detecting cardiac damage, and the current evidence regarding pharmacological cardioprotection. At the end, we focused our attention on the existing guidelines and strategies about the long-term follow-up of childhood cancer survivors.

Are radiation-induced cavernomas clinically relevant findings? Results from long-term follow-up with brain magnetic resonance imaging of childhood cancer survivors.

IntroductionRadiation-induced cavernomas (RIC) after cranial radiotherapy have an unknown risk of hemorrhage. Zabramski magnetic resonance imaging (MRI) classification is touted as being able to indicate non-radiation-induced cavernomas hemorrhage risk. The aim of our study was to assess the hemorrhage risk of RIC during long-term follow-up of childhood cancer survivors based on brain MRI examinations.Patients and methodsWe analyzed retrospectively long-term follow-up data of 36 childhood cancer survivors after initial diagnosis with acute leukemia (n = 18) or brain tumor (n = 18), all treated with cranial radiotherapy. Detected RIC in long-term follow-up brain MRI (1.5 or 3 Tesla) were classified following the Zabramski MRI classification and were categorized into “high” (Zabramski type I, II or V) or “low” (type III or IV) risk of hemorrhage.Results18 patients (50%) showed RIC with a significant relation to the original tumor entity (p = 0.023) and the cumulative radiation dose to the brain (p = 0.016): all 9 childhood cancer survivors diagnosed with medulloblastoma developed RIC. We classified RIC in only 3/36 childhood cancer survivors (8%) (1 patient with acute lymphoblastic leukemia [Zabramski type II] and 2 patients with medulloblastoma [type I and type II]) as high risk for hemorrhage, the remaining RIC were classified as Zabramski type IV with low risk for hemorrhage. None of the childhood cancer survivors with RIC showed symptomatic hemorrhages.ConclusionsRIC are common late effects in childhood cancer survivors treated with cranial radiotherapy affecting half of these patients. However, only a few RIC (occurring in 8% of all reviewed childhood cancer survivors) were classified as high risk for hemorrhage and none of the childhood cancer survivors with RIC developed symptomatic hemorrhages. Thus, we conclude that RIC are low-risk findings in brain MRI and the course is mainly benign.

Is There a Higher Incidence of Sporadic Renal Angiomyolipoma in Childhood Cancer Survivors?

BackgroundCancer treatment can cause various long-term side effects, including those that impact ultrasound findings. During follow-up of childhood cancer survivors (CCSs), we often detected sporadic renal angiomyolipomas without histological confirmation (SAMLs), which is why we initiated this study. We compared the occurrence of SAML in CCSs to the previously reported data from a non-cancer population and correlated SAML with cancer treatment-related factors.MethodsThe cohort included 1098 CCSs (median age at cancer diagnosis (dg) 4.3 years) who had ultrasound follow-up (2014–2019). Of the CCSs, 525 (48%) were female, 132 (12%) had subsequent neoplasms (SNs), and 110 (10%) had genetic syndromes. CCSs were treated for lymphomas 269 (24%) and solid tumors 829 (76%). None of the CCSs had tuberous sclerosis complex (TSC).ResultsSAML developed in 48 (4.4%) CCSs; of these, 20 (42%) had SNs. The coincidence of SAMLs and SNs was found in CCSs with a follow-up period exceeding 20 years. The median age at SAML dg was 27.9 years (interquartile range (IQR) 22.3–34.1), and the median time to SAML dg was 22.6 years (IQR 17.4–27.6). Twenty-one (44%) CCSs developed multiple or bilateral SAMLs lesions; of these, six (12%) were in the radiotherapy field. SAML occurrence correlated with radiotherapy of the retroperitoneum (1.65-fold higher with 95% CI 0.90–3.02). The correlations with other cancer treatment factors and with female sex were less clear.ConclusionThis study revealed the occurrence of SAMLs in CCSs to be 10 times higher than that in non-cancer studies. The current characteristics of CCSs with SAMLs: younger age, and more bilateral or multiple lesions are more similar to TSC associated angiomyolipoma. Moreover, we observed a coincidence of SAMLs with SNs. Our results support the hypothesis that SAML development in CCSs is not simply a late effect of therapy, and indicates other factors are involved in SAML development.

Fertility Status Among Long-Term Childhood Acute Lymphoblastic Leukemia Survivors Enrolled between 1971 and 1998 in the EORTC Children Leukemia Group Trials 58741, 58831/2 and 58881

BackgroundThe survival of childhood acute lymphoblastic leukemia (ALL) has improved significantly over the last decades, with recently reported overall survival rates in Europe of up to 85% in children (<14 years) and 62% in adolescents (15-19 years) (Trama, Lancet Oncol 2016). The increased number of long-term survivors has made the management of side effects an increasingly important issue in pediatric oncology. In this context, the impact on fertility caused by widely employed ALL treatments such as alkylating agents, cranial radiotherapy, total body irradiation and orchiectomy has been an emerging concern over the last years. The aim of this study was to assess fertility among long-term childhood ALL survivors.MethodsBetween 2012 and 2017, a follow-up study was performed among childhood ALL (and acute lymphoblastic lymphoma) survivors enrolled between 1971 and 1998 in EORTC studies (58741, 58831/2, 58881) in France and Belgium. Patients <18 years (yrs) at diagnosis and ≥18 yrs at the time of the follow-up were eligible to fill out a questionnaire that included information about their fertility. The following fertility-related outcomes were evaluated: having menstrual cycles for females, intention to have children, having children, use of reproductive techniques to become pregnant and occurrence of negative pregnancy outcomes (miscarriage, medical abortion, birth defects), separately for males and females.ResultsOut of 1429 eligible patients, 502 (35%) filled out the questionnaire, 862 (60%) were lost to follow-up, and 65 (5%) refused to participate. Disease characteristics and relapse occurrence within one yr after diagnosis were comparable between these 3 groups.Among respondents (275 females, 227 males), approximately 14% were aged 10-17 yrs at diagnosis and almost 30% belonged to the NCI high risk group (Table 1). Approximately 12% received cranial radiotherapy and 6% of females and 8% of males underwent hematopoietic stem cell transplantation. Relapse occurred in 12% and 14% of females and males, respectively. The median time between the diagnosis and the socioeconomic evaluation was 20 yrs (range: 13-42). The median age at follow-up was 25 yrs (range: 18-53).All but 6 females had ever had menstrual cycles. At the time of the fertility evaluation, 93% out of females younger than 45 yrs of age (N=270) either had menstrual cycles, were pregnant, gave breast feeding or continuously used contraceptives. Among females who had tried to become pregnant, 89% had been pregnant, 75% had children and 12% used medical help to become pregnant. One fifth of the ever pregnant females had ever had a miscarriage. The incidences of other negative pregnancy outcomes (e.g. birth defect, medical abortion) were low.The average number of children females had was 0.36 (95% CI: 0.27-0.44). The expected number of children in a population with the same distribution of country, age, and yr of birth of the mothers based on national population registries was 0.55. However, as 52% of the females had a bachelor or master degree and only 3% had no secondary school diploma, this difference could possibly be explained by an overrepresentation of females with a high level of education in our sample.Among males who had tried to have children, 83% made their partner pregnant, 72% had children and 7% used medical help to make their partner pregnant. The incidence of negative pregnancy outcomes other than miscarriage was low.ConclusionsIn this first disease-specific European cohort study, we evaluated fertility among adult survivors of childhood ALL enrolled in EORTC trials, with a median time from the diagnosis of 20 yrs.Most survivors who tried to have children, succeeded (75% of females, 72% of males). However, the average number of children per female seemed to be lower than in the general population. The incidences of birth defects, and medical abortions were low, both among female survivors and the partners of the male survivors.Despite the large number of potentially eligible patients, we could evaluate only 35% of long-term survivors. We were unable to study the effect of specific treatment exposures due to the large variety of treatments that patients received, the association between some treatments (protocol dependent) and age at the fertility evaluation, and the need to stratify the analyses by sex. Improvements in the follow-up of childhood cancer survivors are needed for a better evaluation of the long-term outcomes. [Display omitted] DisclosuresNo relevant conflicts of interest to declare.

Long-Term Follow-Up of Childhood Cancer Survivors and Nurse’s Role

Çocukluk çağı kanserlerinde 5 yıllık sağkalım oranları önemli ölçüde artmış ve sağ kalanların fiziksel ve psikososyal sağlığının sürdürülmesi ön plana çıkmıştır. Sağ kalan çocuk ve adölesanlar uzun dönemde kanserin kendisi ve tedavi maruziyetine bağlı birçok sorunla karşılaşmaktadır. Tedavinin tamamlanmasından yıllar sonra dahi ortaya çıkabilen kardiyak, endokrin, nörolojik, bilişsel, psikolojik ve sosyal geç etkiler sağ kalanların yaşamında önemli kısıtlılıklara neden olabilmektedir. Uzun süreli izlemler ile geç etkilerin önlenmesi ve erken dönemde saptanması, çocuğun fiziksel ve psikososyal gereksinimlerinin karşılanması, çocuk ve ailenin sosyal yaşama uyumu ve çocuğun ikincil risklerden korunması için sağlıklı yaşam tarzının kazandırılması amaçlanmaktadır. İzlemlerin etkin bir şekilde sürdürülmesi için kanıt temelli yaklaşımlara dayandırılması ve multidisipliner ekip tarafından yürütülmesi gerekmektedir. Sağ kalanlara uzun dönemde bütüncül ve aile merkezli bakımın sunulmasında pediatri hemşiresi önemli role sahiptir. Hemşire standart rehberler ışığında oluşturduğu bireyselleştirilmiş bakım planlarıyla sağ kalanların gereksinimlerinin karşılanmasını sağlamaktadır. Bu makalede çocukluk çağı kanserlerinden sağ kalanlarda izlemin önemi, kapsamı ve izlemlerde hemşirenin rolü ele alınmıştır.

What are required for the follow-up of childhood cancer survivors

What are required for the follow-up of childhood cancer survivors

A nationwide questionnaire survey targeting Japanese pediatric endocrinologists regarding transitional care in childhood, adolescent, and young adult cancer survivors.

.Existing guidelines recommend long-term follow-up of childhood cancer survivors (CCS). However, in Japan, transitional care for CCS has not been established. To ascertain the current status in Japan, and to cultivate a better understanding, a questionnaire survey was conducted on transitional care in CCS, and adolescent and young adult (AYA) cancer survivors. Questionnaires were distributed to 183 councilors (137 institutions) of the Japanese Society for Pediatric Endocrinology. A total of 131 responses, representative of 174 councilors, were obtained. The response rate was 95%. Among the respondents, 91% had experience in medical care for cancer patients, while 63% had experience in transitional care; however, the number of patients referred to adult clinics was small. Further, 89% acknowledged the availability of adult endocrinologists who were willing to accept these patients; although their numbers were insufficient. Pediatric endocrinologists highlighted difficulties in medical examinations concerning infertility, obesity, pregnancy/delivery, and gonadal dysfunction, in that order. Staff and time shortages were listed as some of the challenges faced by medical staff, while multisystem morbidity was listed for patients. This nationwide questionnaire survey revealed that Japanese pediatric endocrinologists require cooperation between related departments and collaborative infrastructure to develop transitional care for cancer survivors.

Bone health in childhood cancer: review of the literature and recommendations for the management of bone health in childhood cancer survivors

In the past decades, new cancer treatment approaches for children and adolescents have led to a decrease in recurrence rates and an increase in long-term survival. Recent studies have focused on the evaluation of the late effects on bone of pediatric cancer-related treatments, such as chemotherapy, radiation and surgery. Treatment of childhood cancer can impair the attainment of peak bone mass, predisposing to premature onset of low bone mineral density, or causing other bone side-effects, such as bone quality impairment or avascular necrosis of bone. Lower bone mineral density and microarchitectural deterioration can persist during adulthood, thereby increasing fracture risk. Overall, long-term follow-up of childhood cancer survivors is essential to define specific groups at higher risk of long-term bone complications, identify unrecognized long-term adverse effects, and improve patient care. Children and adolescents with a cancer history should be carefully monitored, and patients should be informed of possible late complications of their previous medical treatment. The International Osteoporosis Foundation convened a working group to review the bone complications of pediatric cancer survivors, outlining recommendations for the management of bone health, in order to prevent and treat these complications.

Cardiovascular Evaluation of Children With Malignancies.

The anthracycline (AC) group of drugs is widely used for cancer chemotherapy and has improved outcomes in many childhood malignancies. However, cardiovascular complications are major causes of morbidity and mortality in AC recipients, with the greatest risk factor being a higher cumulative dosage. The purpose of this review is to describe the etio-pathogenesis and risk factors of AC induced cardiotoxicity, with emphasis on currently available and emerging modalities of non-invasive imaging in its surveillance, and to review guidelines on its prevention and treatment. Presently, ejection fraction and shortening fraction derived from two-dimensional echocardiography are the most widely used parameter for monitoring of cardiac function in childhood cancer survivors. The newer speckle tracking echocardiography has shown potential to detect abnormalities in ventricular function prior to the conventional measures such as ejection fraction and shortening fraction. When available, three-dimensional echocardiography should be used as it allows for more accurate estimation of ejection fraction. Newer magnetic resonance imaging (MRI) techniques, such as delayed enhancement and T1 mapping, are useful adjuncts for cardiac evaluation in cancer survivors, especially in patients with poor echocardiographic windows. Early detection and management of cardiovascular diseases is one of the major goals in the long-term follow-up of childhood cancer survivors. In addition to conventional two-dimensional echocardiography, newer techniques such as speckle tracking echocardiography and three-dimensional echocardiography should be incorporated due to its ability to detect early changes in anthracycline-induced cardiotoxicity. However further research are needed to guide changes in management due to abnormalities in speckle tracking echocardiography.

The hypothalamic-pituitary-adrenal axis in childhood cancer survivors.

Endocrine abnormalities are common among childhood cancer survivors. Abnormalities of the hypothalamic-pituitary-adrenal axis (HPAA) are relatively less common, but the consequences are severe if missed. Patients with tumours located and/or had surgery performed near the hypothalamic-pituitary region and those treated with an accumulative cranial radiotherapy dose of over 30 Gy are most at risk of adrenocorticotrophic hormone (ACTH) deficiency. Primary adrenal insufficiency may occur in patients with tumours located in or involving one or both adrenals. The effects of adjunct therapies also need to be considered, particularly, new immunotherapies. High-dose and/or prolonged courses of glucocorticoid treatment can result in secondary adrenal insufficiency, which may take months to resolve and hence reassessment is important to ensure patients are not left on long-term replacement steroids inappropriately. The prevalence and cumulative incidences of HPAA dysfunction are difficult to quantify because of its non-specific presentation and lack of consensus regarding its investigations. The insulin tolerance test remains the gold standard for the diagnosis of central cortisol deficiency, but due to its risks, alternative methods with reduced diagnostic sensitivities are often used and must be interpreted with caution. ACTH deficiency may develop many years after the completion of oncological treatment alongside other pituitary hormone deficiencies. It is essential that health professionals involved in the long-term follow-up of childhood cancer survivors are aware of individuals at risk of developing HPAA dysfunction and implement appropriate monitoring and treatment.

Parents' preferences for the organisation of long-term follow-up of childhood cancer survivors

Parents' preferences for the organisation of long-term follow-up of childhood cancer survivors

Measuring childhood cancer late effects: evidence of a healthy survivor effect.

Given considerable focus on health outcomes among childhood cancer survivors, we aimed to explore whether survivor bias is apparent during long-term follow-up of childhood cancer survivors. We identified all 1-year survivors of cancer diagnosed before 20years of age in Denmark, Finland, Iceland, and Sweden. From the general population, we randomly sampled a comparison cohort. Study individuals were followed for hospitalizations for diseases of the gastroenterological tract, endocrine system, cardiovascular system, or urinary tract from the start of the cancer registries to 2010. We estimated cumulative incidence with death as competing risk and used threshold regression to compare the hazards of the diseases of interest at ages 20, 40, 60, and 75years. Our study included 27,007 one-year survivors of childhood cancer and 165,620 individuals from the general population. The cumulative incidence of all four outcomes was higher for childhood cancer survivors during early adulthood, but for three outcomes, the cumulative incidence was higher for the general population after age 55years. The hazard ratios (HRs) decreased for all outcomes with increasing age, and for two of the outcomes, the hazards were higher for the general population at older ages (endocrine diseases: age-specific HRs=3.0, 1.4, 1.0, 0.87; Cardiovascular diseases: age-specific HRs=4.1, 1.4, 0.97, 0.84). Our findings provide empirical evidence that survivor bias attenuates measures of association when comparing survivors with the general population. The design and analysis of studies among childhood cancer survivors, particularly as this population attains older ages, should account for survivor bias to avoid misinterpreting estimates of disease burden.

Liver diseases in Adult Life after Childhood Cancer in Scandinavia (ALiCCS): A population-based cohort study of 32,839 one-year survivors.

Information on late onset liver complications after childhood cancer is scarce. To ensure an appropriate follow-up of childhood cancer survivors and reducing late liver complications, the need for comprehensive and accurate information is presented. We evaluate the risk of liver diseases in a large childhood cancer survivor cohort. We included all 1-year survivors of childhood cancer treated in the five Nordic countries. A Cox proportional hazards model was used to estimate hospitalisation rate (hazard) ratios (HRs) for each liver outcome according to type of cancer. We used the risk among survivors of central nervous system tumour as internal reference. With a median follow-up time of 10 years, 659 (2%) survivors had been hospitalised at least once for a liver disease. The risk for hospitalisation for any liver disease was high after hepatic tumour (HR = 6.9) and leukaemia (HR = 1.7). The Danish sub-cohort of leukaemia treated with haematopoietic stem cell transplantation had a substantially higher risk for hospitalisation for all liver diseases combined (HR = 3.8). Viral hepatitis accounted for 286 of 659 hospitalisations corresponding to 43% of all survivors hospitalised for liver disease. The 20-year cumulative risk of viral hepatitis was 1.8% for survivors diagnosed with cancer before 1990 but only 0.3% for those diagnosed after 1990. The risk of liver disease was low but significantly increased among survivors of hepatic tumours and leukaemia. Further studies with focus on the different treatment modalities are needed to further strengthen the prevention of treatment-induced late liver complications.

Parents' preferences for the organisation of long-term follow-up of childhood cancer survivors.

Parents take an important role in follow-up of young cancer survivors. We aimed to investigate (1) parents' preferences for organisation of follow-up (including content, specialists involved and models of care), and (2) parents' and children's characteristics predicting preference for generalist vs. specialist-led follow-up. We sent a questionnaire to parents of childhood cancer survivors aged 11-17years. We assessed on a 4-point Likert scale (1-4), parents' preferences for organisation of long-term follow-up. Proposed models were: telephone/questionnaire, general practitioner (GP) (both categorised as generalist for regression analysis); and paediatric oncologist, medical oncologist or multidisciplinary team (MDT) (categorised as specialists). Of 284 contacted parents, 189 responded (67%). Parents welcomed if visits included checking for cancer recurrence (mean=3.89), late effects screening (mean=3.79), taking patients seriously (mean = 3.86) and competent staff (mean=3.85). The preferred specialists were paediatric oncologists (mean=3.73). Parents valued the paediatric oncologist model of care (mean=3.49) and the MDT model (mean=3.14) highest. Parents of children not attending clinic-based follow-up (OR=2.97, p=.009) and those visiting a generalist (OR=4.23, p=.007) favoured the generalist-led model. Many parents preferred a clinic-based model of follow-up by paediatric oncologists or a MDT. However, parents also valued the follow-up care model according to which their child is followed up.

Childbirth and fertility preservation in childhood and adolescent cancer patients: a second national survey of Japanese pediatric endocrinologists.

.Although existing guidelines recommend long-term follow-up of childhood cancer survivors (CCSs), their fertility has not been fully investigated in Japan. To address this issue, we organized a working panel consisting of medical specialists in foundation hospitals. We conducted questionnaire surveys targeting pediatric endocrinologists regarding reproduction in pediatric and adolescent cancer patients in collaboration with the CCS committee of the Japanese Society for Pediatric Endocrinology (JSPE). The first questionnaire was sent to 178 directors or councilors of the JSPE, and the second was sent to those who had provided answers on their experience with childbirth or fertility preservation. A total of 151 responses (84.8%) were obtained in the first survey. In the second survey, the response rate was 100% (39 respondents). There were 27 answers describing experiences with childbirth (16 from partners of male CCSs, 22 from female CCSs). A few cases of premature birth and low birth weight were reported. There were 25 answers describing experiences with fertility preservation; 21 were from male and 17 from female CCSs. It was mainly physicians who recommended fertility preservation. This nationwide questionnaire survey revealed that a limited number of Japanese pediatric endocrinologists had experience with childbirth and fertility preservation in CCSs. A further long-term follow-up study of their fertility is needed.

Late Cardiac Events after Childhood Cancer: Methodological Aspects of the Pan-European Study PanCareSurFup.

Background and AimChildhood cancer survivors are at high risk of long-term adverse effects of cancer and its treatment, including cardiac events. The pan-European PanCareSurFup study determined the incidence and risk factors for cardiac events among childhood cancer survivors. The aim of this article is to describe the methodology of the cardiac cohort and nested case-control study within PanCareSurFup.MethodsEight data providers in Europe participating in PanCareSurFup identified and validated symptomatic cardiac events in their cohorts of childhood cancer survivors. Data on symptomatic heart failure, ischemia, pericarditis, valvular disease and arrhythmia were collected and graded according to the Criteria for Adverse Events. Detailed treatment data, data on potential confounders, lifestyle related risk factors and general health problems were collected.ResultsThe PanCareSurFup cardiac cohort consisted of 59,915 5-year childhood cancer survivors with malignancies diagnosed between 1940 and 2009 and classified according to the International Classification of Childhood Cancer 3. Different strategies were used to identify cardiac events such as record linkage to population/ hospital or regional based databases, and patient- and general practitioner-based questionnaires.ConclusionThe cardiac study of the European collaborative research project PanCareSurFup will provide the largest cohort of 5-year childhood cancer survivors with systematically ascertained and validated data on symptomatic cardiac events. The result of this study can provide information to minimize the burden of cardiac events in childhood cancer survivors by tailoring the follow-up of childhood cancer survivors at high risk of cardiac adverse events, transferring this knowledge into evidence-based clinical practice guidelines and providing a platform for future research studies in childhood cancer patients.

Gonadal function, fertility, and reproductive medicine in childhood and adolescent cancer patients: a national survey of Japanese pediatric endocrinologists.

.An increasing number of pediatric cancer patients survive, and treatment-related infertility represents one of the most important issues for these patients. While official guidelines in Japan recommend long-term follow-up of childhood cancer survivors (CCSs), their gonadal function and fertility have not been clarified. To address this issue, we organized a working panel to compile evidence from long-term survivors who received treatments for cancer during childhood or adolescence. In collaboration with members of the CCS Committee of the Japanese Society for Pediatric Endocrinology (JSPE), we conducted a questionnaire survey regarding reproductive function in pediatric cancer patients. A cross-sectional survey was sent to 178 JSPE-certified councilors who were asked to self-evaluate the medical examinations they had performed. A total of 151 responses were obtained, revealing that 143 endocrinologists were involved in the care of CCSs. A quarter of the respondents reported having experienced issues during gonadal or reproductive examinations. Several survivors did not remember or fully understand the explanation regarding gonadal damage, and faced physical and psychological distress when discussing the risk of becoming infertile. Pediatric endocrinologists had anxieties regarding their patients’ infertility and the risk of miscarriage, premature birth, and delivery problems. Only a limited number of endocrinologists had experience with managing childbirth and fertility preservation. Many councilors mentioned the necessity for inter-disciplinary communication among healthcare providers. Both endocrinologists and oncologists should set and follow a uniform clinical guideline that includes management of fertility of CCSs.

Uncovering healthcare practitioners’ information processing using the think-aloud method: From paper-based guideline to clinical decision support system

ObjectiveTo investigate whether the use of the think-aloud method with propositional analysis could be helpful in the design of a Clinical Decision Support System (CDSS) providing guideline recommendations about long-term follow-up of childhood cancer survivors. Materials and methodsThe think-aloud method was used to gain insight into healthcare professionals’ information processing while reviewing a paper-based guideline. A total of 13 healthcare professionals (6 physicians and 7 physician assistants) prepared 2 fictitious patient consults using the paper-based guideline. Propositional analysis was used to analyze verbal protocols of the think-aloud sessions. A prototype CDSS was developed and a usability study was performed, again with the think-aloud method. ResultsThe analysis revealed that the paper-based guideline did not support healthcare practitioners in finding patient-specific recommendations. An information processing model for retrieving recommendations was developed and used as input for the design of a CDSS prototype user interface. Usability analysis of the prototype CDSS showed that the navigational structure of the system fitted well with healthcare practitioners’ daily practices. ConclusionsThe think-aloud method combined with propositional analysis of healthcare practitioners’ verbal utterances while they processed a paper-based guideline was useful in the design of a usable CDSS providing patient-specific guideline recommendations.