18 research outputs found
Health‐related quality of life of children born to childhood cancer survivors in Germany
Objective: Rising childhood cancer survival rates have increased the importance of health-related quality of life (HRQL) assessment. While survivors show comparable HRQL to peers, concerns that cancer treatment could impact the health of prospective children were reported. No previous publications address HRQL of childhood cancer survivor offspring.
Methods: We assessed survivor offspring HRQL using the parental KINDL questionnaire. Matched-pair analysis was conducted with data from the general population (KiGGS study) using age, gender and education (1:1, n = 1206 cases). Multivariate analyses were conducted to detect the influence of parental diagnose and treatment on offspring HRQL.
Results: Overall, within KINDL dimensions, survivors reported comparable to higher HRQL for their children than the general population. Survivor parents reported significantly (p < 0.001) higher psychological (86.7% vs. 83.0%, Cohen's d = 0.3) and self-esteem (79.1% vs. 73.3%, Cohen's d = 0.5) well-being scores for younger children (3-6-year-olds). As time since diagnosis increased, parents reported higher well-being scores. Accordingly, recently diagnosed survivors reported significantly lower psychological well-being scores (p = 0.28; OR = 0.457; 95% CI = 0.228-0.918) for their children. With increasing age, average HRQL scores decreased in both cohorts; yet, this drop was less pronounced for survivor offspring. The biggest difference between age groups (7-10- vs. 14-17-year-olds) was found for school-specific well-being (6.2-point drop in survivor offspring vs. 18.2-point drop in KiGGS offspring).
Conclusion: Comparable to higher parentally assessed HRQL was reported for survivor offspring compared to peers. These findings are reassuring and consistent with self-reported HRQL in childhood cancer survivors. Type of parental cancer diagnosis and treatment showed no negative impact on offspring HRQL
Health‐relatedquality of life of children born to childhood cancer survivors in Germany
Objective:
Rising childhood cancer survival rates have increased the importance of health-related quality of life (HRQL) assessment. While survivors show comparable HRQL to peers, concerns that cancer treatment could impact the health of prospective children were reported. No previous publications address HRQL of childhood cancer survivor offspring.
Methods:
We assessed survivor offspring HRQL using the parental KINDL questionnaire. Matched-pair analysis was conducted with data from the general population (KiGGS study) using age, gender and education (1:1, n = 1206 cases). Multivariate analyses were conducted to detect the influence of parental diagnose and treatment on offspring HRQL.
Results:
Overall, within KINDL dimensions, survivors reported comparable to higher HRQL for their children than the general population. Survivor parents reported significantly (p < 0.001) higher psychological (86.7% vs. 83.0%, Cohen's d = 0.3) and self-esteem (79.1% vs. 73.3%, Cohen's d = 0.5) well-being scores for younger children (3–6-year-olds). As time since diagnosis increased, parents reported higher well-being scores. Accordingly, recently diagnosed survivors reported significantly lower psychological well-being scores (p = 0.28; OR = 0.457; 95% CI = 0.228–0.918) for their children. With increasing age, average HRQL scores decreased in both cohorts; yet, this drop was less pronounced for survivor offspring. The biggest difference between age groups (7–10- vs. 14–17-year-olds) was found for school-specific well-being (6.2-point drop in survivor offspring vs. 18.2-point drop in KiGGS offspring).
Conclusion:
Comparable to higher parentally assessed HRQL was reported for survivor offspring compared to peers. These findings are reassuring and consistent with self-reported HRQL in childhood cancer survivors. Type of parental cancer diagnosis and treatment showed no negative impact on offspring HRQL.Peer Reviewe
Health outcomes in offspring born to survivors of childhood cancers following assisted reproductive technologies
Purpose: An increasing number of childhood cancer survivors are using assisted reproductive technologies (ART) to overcome treatment-related fertility impairment. We report perinatal and health outcomes of offspring born to survivors following ART.
Methods: The FeCt Multicenter Offspring Study surveyed the health of offspring of childhood cancer survivors. Health outcomes in offspring born to survivors following ART (n = 57, 4.6%) or after spontaneous conception (n = 1182) were assessed in the German cohort (n = 1239) using bivariate analysis. Findings were put into the context of the general German population by health outcome assessment in 1:1 matched-pair analysis (n = 2478).
Results: Nearly twice the survivors used ART compared with numbers reported for the German general population (4.6% vs. 2.6%). Successful pregnancies were achieved after a median of two cycles, mainly using non-cryopreserved oocytes/sperm. Multiple sibling births (p < 0.001, 28.1% vs. 3.0%) and low birth weight (p = 0.008; OR = 2.659, 95% CI = 1.258-5.621) occurred significantly more often in offspring born to survivors who utilized ART than spontaneously conceived children, whereas similar percentages were born preterm or too small for their gestational age. ART did not increase the prevalence of childhood cancer or congenital malformations in offspring born to survivors.
Conclusion: ART use by childhood cancer survivors was successful with both fresh and cryopreserved oocytes/sperm, and did not influence perinatal health or health outcomes when known confounders were taken into account.
Implications for cancer survivors: Oncofertility is an important component of patient care. Our study implicates that the utilization of ART by adult survivors of childhood cancer does not put offspring at additional risk for adverse perinatal or health outcomes
The Use of Assisted Reproductive Technology by European Childhood Cancer Survivors
CCS often wish to have biological children yet harbour concerns about fertility impairment, pregnancy risks and the general health risks of prospective offspring. To clarify these concerns, health outcomes in survivor offspring born following ART (n = 74, 4.5%) or after spontaneous conception (n = 1585) were assessed in our European offspring study by descriptive and bivariate analysis. Outcomes were compared to a sibling offspring cohort (n = 387) in a 4:1 matched-pair analysis (n = 1681). (i) Survivors were more likely to employ ART than their siblings (4.5% vs. 3.7%, p = 0.501). Successful pregnancies were achieved after a median of one cycle with, most commonly, intracytoplasmic sperm injection (ICSI) using non-cryopreserved oocytes/sperm. (ii) Multiple-sibling births (p < 0.001, 29.7% vs. 2.5%), low birth weight (p < 0.001; OR = 3.035, 95%-CI = 1.615-5.706), and preterm birth (p < 0.001; OR = 2.499, 95%-CI = 1.401-4.459) occurred significantly more often in survivor offspring following ART utilisation than in spontaneously conceived children. ART did not increase the prevalence of childhood cancer, congenital malformations or heart defects. (iii) These outcomes had similar prevalences in the sibling population. In our explorative study, we could not detect an influence on health outcomes when known confounders, such as multiple births, were taken into account
Fertility education for adolescent cancer patients: Gaps in current clinical practice in Europe
Objective:
As adolescent cancer patients may suffer from infertility following treatment, fertility counselling is essential. Our aim was to explore the current situation in four European countries in terms of (I) education about the risk for infertility, (II) counselling on fertility preservation, (III) patients' knowledge on fertility, (IV) sufficiency of information and (V) uptake of cryopreservation.
Methods:
In total, 113 patients (13–20 years) at 11 study centres completed a self-report questionnaire three and six months after cancer diagnosis. Multivariate logistic regression was used to estimate odds ratios (OR) with 95% confidence intervals (CI).
Results:
As many as 80.2% of participants reported having received education about the risk for infertility prior to treatment, 73.2% recalled counselling on fertility preservation. Only 52.3% stated they felt sufficiently informed to make a decision. Inability to recall counselling on fertility preservation (OR = 0.03, CI: 0.00–0.47) and female gender (OR = 0.11, CI: 0.03–0.48) was associated with lower use of cryopreservation, whereas older age was associated with higher use.
Conclusion:
Fertility counselling was available to a relatively high proportion of patients, and it did influence the utilisation of cryopreservation. However, many patients did not feel sufficiently informed. Further improvement is needed to enable adolescent cancer patients to make an informed decision on fertility preservation
Desire for biological parenthood and patient counseling on the risk of infertility among adolescents and adults with hemoglobinopathies.
BACKGROUND
Both diagnosis and treatment of hemoglobinopathies have been associated with an increased risk of fertility impairment. German guidelines recommend annual monitoring of fertility parameters to enable early detection of fertility impairment and/or to offer fertility preservation (FP) when indicated. We explored the general desire for parenthood, the frequency of recalling fertility counseling and testing, and the utilization of FP in adolescents and adults with hemoglobinopathies.
PROCEDURE
In a cross-sectional study, patients aged 12-50 years, treated in Germany, Austria, or Switzerland, were surveyed on fertility-related aspects. Medical data, including fertility testing results, were collected from patient records.
RESULTS
Overall, 116/121 eligible patients, diagnosed with sickle cell disease (70.7%), thalassemia (27.6%), or other hemoglobinopathy (1.7%), participated in our study (57.8% female, median age 17.0 years, range 12-50 years). All participants required treatment of the underlying hemoglobinopathy: 68.1% received hydroxyurea, 25.9% required regular blood transfusions, and 6.0% underwent hematopoietic stem cell transplantation (HSCT). Most patients (82/108, 75.9%) stated a considerable to strong desire for (future) parenthood, independent of sex, education, diagnosis, or subjective health status. Fertility counseling was only recalled by 32/111 patients (28.8%) and least frequently by younger patients (12-16 years) or those treated with regular blood transfusions or hydroxyurea. While fertility testing was documented for 59.5% (69/116) in medical records, only 11.6% (13/112) recalled previous assessments. FP was only used by 5.4% (6/111) of patients.
CONCLUSION
Most patients with hemoglobinopathies wish to have biological children, yet only few recalled fertility counseling and testing. Adequate patient counseling should be offered to all patients at risk for infertility
Recommendations for gonadotoxicity surveillance in male childhood, adolescent, and young adult cancer survivors : a report from the International Late Effects of Childhood Cancer Guideline Harmonization Group in collaboration with the PanCareSurFup Consortium
Treatment with chemotherapy, radiotherapy, or surgery that involves reproductive organs can cause impaired spermatogenesis, testosterone deficiency, and physical sexual dysfunction in male pubertal, adolescent, and young adult cancer survivors. Guidelines for surveillance and management of potential adverse effects could improve cancer survivors' health and quality of life. Surveillance recommendations vary considerably, causing uncertainty about optimum screening practices. This clinical practice guideline recommended by the International Late Effects of Childhood Cancer Guideline Harmonization Group in collaboration with the PanCareSurFup Consortium, developed using evidence-based methodology, critically synthesises surveillance recommendations for gonadotoxicity in male childhood, adolescent, and young adult (CAYA) cancer survivors. The recommendations were developed by an international multidisciplinary panel including 25 experts in relevant medical specialties, using a consistent and transparent process. Recommendations were graded according to the strength of underlying evidence and potential benefit gained by early detection and appropriate management. The aim of the recommendations is to enhance evidence-based care for male CAYA cancer survivors. The guidelines reveal the paucity of high-quality evidence, highlighting the need for further targeted research.Peer reviewe
Health-related quality of life in European childhood cancer survivors: Protocol for a study within PanCareLIFE
Background: Survival after childhood cancer has improved to more than 80% during the last few years, leading to an increased number of childhood cancer survivors. Cancer itself, or its treatment, may cause chronic health conditions, including somatic and mental sequelae, which may affect survivors’ health-related quality of life (HRQoL). Objective: The project PanCareLIFE aims to establish a large database with comprehensive data on childhood cancer survivors from different European countries, including data on HRQoL. Within PanCareLIFE, this study aims to describe HRQoL in survivors, investigate predictors of HRQoL, and describe the association of HRQoL with hearing and female fertility impairment. This paper describes the design of the HRQoL study, the origin of data, strategies for data collection, and sampling characteristics of survivors from each contributing country. Methods: A total of 6 institutions from 5 European countries (the Czech Republic, France, Germany, the Netherlands, and Switzerland) provided data on HRQoL assessed with the Short Form 36 and on relevant predictors. The central PanCareLIFE data center aggregated the data and harmonized the variables between the institutions. Survivors were eligible if they received a diagnosis of cancer according to the 12 main groups of the International Classification of Childhood Cancer, 3rd edition, or Langerhans cell histiocytosis; were aged ≤18 years at the time of diagnosis; were residents of the respective country at the time of diagnosis; had survived ≥5 years after cancer diagnosis; were aged ≥18 years at the time of the questionnaire survey; and did not refuse to registration in the national or local childhood cancer cohort. Results: We identified 24,993 eligible survivors. Of those, 19,268 survivors received a questionnaire and 9871 survivors participated, resulting in response rates of 9871/24,993 (39.50%) of eligible survivors and of 9871/19,268 (51.23%) invited survivors. Most participants were diagnosed with cancer between the ages of 10 and 14 years (3448/9871, 34.93%) or <5 years (3201/9871, 32.43%). The median age was 8 years. Of the 9871 participants, 3157 (31.97%) were survivors of leukemia, 2075 (21.02%) lymphoma, and 1356 (13.7%) central nervous system (CNS) tumors. Most participants (9225/9871, 93.46%) had no history of a subsequent tumor; 77.45% (7645/9871) received chemotherapy with or without other treatments. More than half (5460/9871, 55.31%) were aged 25 to 34 years at the time of the HRQoL study. Participating survivors differed from nonparticipants; participants were more often women, survivors of leukemia or lymphoma, and less frequently, survivors of CNS tumors than nonparticipants. Conclusions: PanCareLIFE successfully assessed HRQoL and its predictors in 9871 European survivors of childhood cancer. This large population will permit detailed investigations of HRQoL after childhood cancer, particularly the impact of hearing and female fertility impairment on HRQoL
Stem cell transplantation in childhood acute lymphoblastic leukemia
Ziel der vorliegenden Habilitationsschrift war es, die Rolle der SZT bei der
Behandlung von Kindern und Jugendlichen mit Rezidiv einer ALL zu
spezifizieren. Hier war es zunächst wichtig im Rahmen der ALL Rezidiv Studien
zu klären, welche therapeutische Effizienz verschiedene Behandlungstrategien
d.h. Chemotherapie vs allogener vs autologer SZT bei extramedullärem Rezidiv
einer ALL in 2. oder nachfolgender Remission haben. Bei dieser Patientengruppe
handelt es sich um Patienten die eine relativ gute Prognose mit
konventioneller Chemo- und Strahlentherapie haben. Es war im Weiteren von
Interesse, ob es bei Patienten mit spätem Knochenmarkrezidiv möglich ist, in
2. CR zunächst keine allogene SZT- zumindest keine von einem unverwandten
Spender- durchzuführen und diese ggf. in 3. CR nachzuholen, um so einigen
Patienten die therapiebedingte Toxizität, mit der allogene SZT von
unverwandten Spendern einhergehen, zu ersparen. Bei dieser Patientengruppe
handelt es sich um Patienten die eine intermediäre Prognose mit
konventioneller Chemo- und Strahlentherapie haben. Für Patienten ohne
geeigneten Stammzellspender ist die allogene SZT nicht anwendbar. Für Kinder
der Hochrisikogruppe wäre prinzipiell die autologe Stammzelltransplantation
mit nachfolgender Immuntherapie nach Hochdosistherapie eine therapeutische
Alternative. Zunächst könnte die überwiegende Tumormasse durch
Chemoradiotherapie vernichtet werden. Nach Stammzellreinfusion könnten dann
körpereigene immunologische Abwehrmechanismen stimuliert werden, die zu einer
stabilen Remission führen. Um dieses zu erreichen, wurde ein
Vakzinationsverfahren entwickelt, bei dem Leukämiezellen durch Einschleusen
einer für ein fremdes HLA codierenden cDNA allogenisiert werden. In der
vorliegenden Habilitationsschrift wird evaluiert, ob die autologe SZT in ihrer
bisherigen Form d.h. ohne nachfolgende Immun- oder Dauertherapie einen
prognostischen Vorteil gegenüber der konventionellen Chemotherapie für Kinder
mit Rezidiv einer akuten lymphoblastischen Leukämie erbringt. Auch wird
evaluiert, wo die SZT von unverwandten Spendern mit ihren noch immer
beträchtlichen Mortalitäts und Morbiditätsrisiken ihre Berechtigung findet.
D.h. welche Patienten ein so hohes Rezidiv Risiko haben, dass eine
Fremdspender SZT indiziert ist und umgekehrt welche Patienten sie für eine
Langzeit Kontrolle Ihrer Leukämie nicht benötigen. Es werden die Auswirkungen
der Ganzkörperbestrahlung im Rahmen der Konditionierung vor SZT auf die akute
Toxizität bei Kindern mit malignen Erkrankungen dargestellt. Es wird das
Risiko geschätzt, nach ALL- Erst- und Rezidivtherapie eine maligne
Zweiterkrankung zu erleiden.The Aim of this professorial dissertation was to specify the role of stem cell
transplantation (SCT) for the treatment of relapsed acute lymphoblastic
leukemia (ALL) in childhood and adolescence. Herefore, it was important to
clarify in the context of the ALL relapse studies, which therapeutical
efficacy different treatment strategies i.e. chemotherapy vs allogeneic vs
autologous SCT have for isolated extramedullary relapse in children with ALL
in second or following remission. This group of patients has a relatively good
prognosis with conventional chemo- and radiotherapy alone. It was of interest
that allogeneic SCT is a conceivable alternative for a subset of children with
ALL in a third remission. This group of patients has an intermediate prognosis
with conventional chemo- and radiotherapy. For patients without a suitable
donor allogeneic SCT is not applicable. For children with high risk of relapse
autologous SCT followed by immunotherapy could be a therapeutical alternative.
Therefore, we developed a vaccination with autologous leukemic cells
transfected with a cDNA expression plasmid coding for an allogeneic HLA class
I antigen combined with interleukin-2 treatment. Conventional autologous SCT
was compared with chemotherapy for children with ALL in a second remission by
matched pair analysis. Unrelated donor stem cell transplantation was compared
with chemotherapy for children with ALL in a second remission by matched-pair
analysis. The influence of fractionated total body irradiation on mucosal
toxicity in intensified conditioning regimens for autologous SCT in pediatric
cancer patients was shown. Secondary malignant neoplasms after intensive
treatment of relapsed ALL in childhood were evaluated and discussed. The
outcome of these studies contribute to clarification as to which patients are
at such high risk of recurrence that allogeneic SCT is indicated and,
conversely, which patients do not require transplantation for long-term
disease control