Childhood and Adolescent Cancer

A special issue of Cancers (ISSN 2072-6694). This special issue belongs to the section "Pediatric Oncology".

Deadline for manuscript submissions: closed (31 March 2022) | Viewed by 39530

Special Issue Editors


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Guest Editor
1. Childhood Cancer Epidemiology and Outcomes, Princess Máxima Center for Pediatric Oncology, 3015 GD Utrecht, The Netherlands
2. Department Research and Development, Netherlands Comprehensive Cancer Organisation (IKNL), 3511 DT Utrecht, The Netherlands
Interests: AYA oncology; cancer surveillance; epidemiology; pediatric oncology; survival disparities; time trends

E-Mail Website
Guest Editor
Princess Máxima Center for Pediatric Oncology, Utrecht, The Netherlands
Interests: childhood acute lymphoblastic leukemia (ALL); childhood and adolescent cancer; pediatric oncology

Special Issue Information

Dear Colleagues,  

Cancer is a relatively rare disease in children and adolescents; however, cancer incidence is increasing slightly over time. Fortunately, 5-year survival has improved from about 40% in the 1960s to more than 80% today in high-income countries. This survival improvement is the result of improved diagnostics, development of effective therapies, improved supportive care, and centralization of care. However, a variety of cancers in adolescents have poorer survival outcomes than the same cancers in children. Many factors have been identified which possibly contribute to this survival disparity, including differences in disease characteristics, diagnostic process, clinical trial participation, treatment strategies, adherence to treatment, and treatment-related toxicities.

The focus of this Special Issue is on highlighting studies on differences between children and adolescents considering the following aspects: (a) incidence, survival, and mortality; (b) disease biology; (c) diagnosis; (d) clinical trial participation; (e) site of treatment; (f) treatment strategies; (g) treatment-related toxicities; (h) survivorship care; (i) palliative care.

Dr. Henrike Karim-Kos
Dr. Rob Pieters
Guest Editors

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Keywords

  • cancer in children and adolescents
  • incidence, survival and mortality
  • disease biology
  • diagnosis
  • clinical trial participation
  • site of treatment
  • treatment strategies
  • treatment-related toxicities
  • survivorship care
  • palliative care

Published Papers (13 papers)

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Research

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14 pages, 537 KiB  
Article
Prevalence and Risk Factors for Hyposalivation and Xerostomia in Childhood Cancer Survivors Following Different Treatment Modalities—A Dutch Childhood Cancer Survivor Study Late Effects 2 Clinical Study (DCCSS LATER 2)
by Juliette Stolze, Jop C. Teepen, Judith E. Raber-Durlacher, Jacqueline J. Loonen, Judith L. Kok, Wim J. E. Tissing, Andrica C. H. de Vries, Sebastian J. C. M. M. Neggers, Eline van Dulmen-den Broeder, Marry M. van den Heuvel-Eibrink, Helena J. H. van der Pal, A. Birgitta Versluys, Margriet van der Heiden-van der Loo, Marloes Louwerens, Leontien C. M. Kremer, Henk S. Brand and Dorine Bresters
Cancers 2022, 14(14), 3379; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14143379 - 11 Jul 2022
Cited by 7 | Viewed by 1893
Abstract
Background: Limited data are available on the risk factors of salivary gland dysfunction in long-term childhood cancer survivors (CCS). The objective of this cross-sectional study, part of the multidisciplinary multicenter Dutch CCS Study Late Effects 2 (DCCSS LATER 2), was to assess the [...] Read more.
Background: Limited data are available on the risk factors of salivary gland dysfunction in long-term childhood cancer survivors (CCS). The objective of this cross-sectional study, part of the multidisciplinary multicenter Dutch CCS Study Late Effects 2 (DCCSS LATER 2), was to assess the prevalence of and risk factors for hyposalivation and xerostomia in CCS. Methods: From February 2016 until March 2020, 292 CCS were included. Data with regard to gender, age at study, diagnosis, age at diagnosis, and treatment characteristics were collected, as well as the unstimulated (UWS) and stimulated whole salivary flow rate (SWS). Xerostomia was assessed with the Xerostomia Inventory (XI) questionnaire. Multivariable Poisson regression analyses were used to evaluate the association between potential risk factors and the occurrence of hyposalivation. Results: The minimum time between diagnosis and study enrollment was 15 years. The prevalence of hyposalivation was 32% and the prevalence of xerostomia was 9.4%. Hyposalivation and xerostomia were not significantly correlated. Risk factors for hyposalivation were female gender and a higher dose of radiotherapy (>12 Gy) to the salivary gland region. Conclusion: Considering the importance of saliva for oral health, screening for hyposalivation in CCS is suggested in order to provide optimal oral supportive care aimed to improve oral health. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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14 pages, 326 KiB  
Article
Self-Reported Health Problems and Quality of Life in a Sample of Colombian Childhood Cancer Survivors: A Descriptive Cross-Sectional Study
by Natalia Godoy-Casasbuenas and Esther de Vries
Cancers 2022, 14(12), 2999; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14122999 - 18 Jun 2022
Cited by 1 | Viewed by 2075
Abstract
Objectives: To describe the self-reported health problems and quality of life among adult-aged Colombian childhood and adolescent cancer survivors. Methods: This is a descriptive cross-sectional study with Colombian childhood cancer survivors (CCS) who were diagnosed before the age of 18, at the moment [...] Read more.
Objectives: To describe the self-reported health problems and quality of life among adult-aged Colombian childhood and adolescent cancer survivors. Methods: This is a descriptive cross-sectional study with Colombian childhood cancer survivors (CCS) who were diagnosed before the age of 18, at the moment of study were ≥18 years, and at least 5 years had passed since diagnosis. Each participant completed a self-reported study-specific online questionnaire along with the SF-36 Health Survey to assess the prevalence of health problems and current quality of life (QoL). Data were analyzed using descriptive statistics and independent sample t-tests. Results: Out of the 122 CCS who participated, 100% reported at least one health problem, mostly gastritis, headaches, and lack of concentration, followed by obesity and fertility issues. In general, they had a good perception of their QoL, which was, on average, only diminished in the areas of vitality, emotional role functioning, and social functioning. Conclusion: Perceived health problems among the participating Colombian CCS were prevalent; most reported a good self-perceived QoL. This is the first study on understanding health problems and QoL of CCS treated in Colombia and South America. It reopens the debate on the need to carry out long-term follow-up in this population among Colombian society. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
11 pages, 460 KiB  
Article
Effect of Treatment in a Specialized Pediatric Hemato-Oncology Setting on 5-Year Survival in Acute Lymphoblastic Leukemia: A Quasi-Experimental Study
by Margrietha van der Linde, Nikki van Leeuwen, Frank Eijkenaar, Anita W. Rijneveld, Rob Pieters and Henrike E. Karim-Kos
Cancers 2022, 14(10), 2451; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14102451 - 16 May 2022
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Abstract
Survival rates of adolescents and young adults (AYAs) with acute lymphoblastic leukemia (ALL) are inferior to those of pediatric ALL patients. In part, this may be caused by differences in treatment setting. Generally, children are treated in specialized pediatric hemato-oncology settings, whereas AYAs [...] Read more.
Survival rates of adolescents and young adults (AYAs) with acute lymphoblastic leukemia (ALL) are inferior to those of pediatric ALL patients. In part, this may be caused by differences in treatment setting. Generally, children are treated in specialized pediatric hemato-oncology settings, whereas AYAs are treated in adult hemato-oncology settings. Since 2005, adult treatment protocols have included pediatric-inspired chemotherapy, which has been the standard of care for AYAs from 2008 onwards. This study aims to assess whether, despite protocols in both settings having become more similar, there remains an effect of treatment in specialized pediatric hemato-oncology settings on 5-year survival for ALL patients in the Netherlands. We used nationwide registry data (2004–2013) on 472 ALL patients aged between 10 and 30 years old. A fuzzy regression discontinuity design was applied to estimate the treatment effect using two-stage least squares regression with the treatment threshold at 17 years and 7 months of age, adjusting for sex, age at diagnosis, and immunophenotype. We found a risk difference of 0.419 (p = 0.092; 95% CI = −0.0686; 0.907), meaning a 41.9 percentage point greater probability of surviving five years after diagnosis for ALL patients treated in specialized pediatric hemato-oncology settings. Our results suggest that ALL patients around the threshold could benefit from increased collaboration between pediatric and adult hemato-oncology in terms of survival. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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11 pages, 1675 KiB  
Article
The Cytogenetic Landscape of Pediatric Chronic Myeloid Leukemia Diagnosed in Chronic Phase
by Axel Karow, Gudrun Göhring, Stephanie Sembill, Friederike Lutterloh, Fina Neuhaus, Sara Callies, Elke Schirmer, Zofia Wotschofsky, Oisin Roche-Lancaster, Meinolf Suttorp, Manuela Krumbholz and Markus Metzler
Cancers 2022, 14(7), 1712; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14071712 - 28 Mar 2022
Cited by 6 | Viewed by 1971
Abstract
Philadelphia chromosome-positive chronic myeloid leukemia (CML) is cytogenetically characterized by the classic translocation t(9;22)(q34;q11), whereas additional non-Philadelphia aberrations (nPhAs) have been studied extensively in adult patients with CML, knowledge on nPhAs in pediatric patients with CML is still sparse. Here, we have determined [...] Read more.
Philadelphia chromosome-positive chronic myeloid leukemia (CML) is cytogenetically characterized by the classic translocation t(9;22)(q34;q11), whereas additional non-Philadelphia aberrations (nPhAs) have been studied extensively in adult patients with CML, knowledge on nPhAs in pediatric patients with CML is still sparse. Here, we have determined nPhAs in a cohort of 161 patients younger than 18 years diagnosed with chronic phase CML and consecutively enrolled in the German national CML-PAED-II registry. In 150 cases (93%), an informative cytogenetic analysis had been performed at diagnosis. In total, 21 individuals (13%) showed nPhAs. Of these, 12 (8%) had a variant translocation, 4 (3%) additional chromosomal aberrations (ACAs) and 5 (3%) harbored a complex karyotype. Chromosome 15 was recurrently involved in variant translocations. No significant impact of the cytogenetic subgroup on the time point of cytogenetic response was observed. Patients with a complex karyotype showed an inferior molecular response compared to patients carrying the classic translocation t(9;22)(q34;q11), variant translocations or ACAs. No significant differences in the probability of progression-free survival and overall survival was found between patients with nPhAs and patients with the classic Philadelphia translocation only. Our results highlight the distinct biology of pediatric CML and underline the need for joint international efforts to acquire more data on the disease pathogenesis in this age group. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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12 pages, 958 KiB  
Article
Pain Coping Strategies in Pediatric Patients with Acute Leukemias in the First Month of Therapy: Effects of Treatments and Implications on Procedural Analgesia
by Marta Tremolada, Giulia Tasso, Roberta Maria Incardona, Manuela Tumino, Maria Caterina Putti, Alessandra Biffi and Marta Pillon
Cancers 2022, 14(6), 1473; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14061473 - 14 Mar 2022
Cited by 1 | Viewed by 2125
Abstract
Children with leukemia experience difficulties adapting to medical procedures and to the chemotherapy’s adverse effects. Study’s objectives were to identify which coping strategies could be associated with the treatments’ factors and with the dosage of sedation analgesic drugs during bone marrow aspirates. A [...] Read more.
Children with leukemia experience difficulties adapting to medical procedures and to the chemotherapy’s adverse effects. Study’s objectives were to identify which coping strategies could be associated with the treatments’ factors and with the dosage of sedation analgesic drugs during bone marrow aspirates. A total of 125 patients (mean = 6.79 years; standard deviation = 3.40), majority with acute lymphoblastic leukemia (90.4%) and their parents received, one month after diagnosis, the Pediatric Pain Coping Inventory. Data on the severe treatment effects and on the dosage of drugs in sedation-analgesia were also collected. An ANCOVA model (R2 = 0.25) showed that, weighing the age factor (F = 3.47; df = 3; p = 0.02), the number of episodes of fever (F = 4.78; df = 1; p = 0.03), nausea (F = 4.71; df = 1; p = 0.03) and mucositis (F = 5.81; df = 1; p = 0.02) influenced the use of distraction. Cognitive self-instructions (R2 = 0.22) were influenced by the number of hospitalizations (F = 5.14; df = 1; p = 0.03) and mucositis (F = 8.48; df = 3; p = 0.004) and by child’s age (F = 3.76; df = 3; p = 0.01). Children who sought parental support more frequently (F = 9.7; df = 2; p = 0.0001) and who tended not to succumb to a catastrophic attitude (F = 13.33; df = 2; p = 0.001) during the induction treatment phase required lower drug dosages, especially propofol. The clinical application of these results could be to encourage the use of cognitive self-instructions and search for social support. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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16 pages, 1098 KiB  
Article
Outcome of Children and Adolescents with Recurrent Classical Hodgkin Lymphoma: The Italian Experience
by Alberto Garaventa, Stefano Parodi, Giulia Guerrini, Piero Farruggia, Alessandra Sala, Marta Pillon, Salvatore Buffardi, Francesca Rossi, Maurizio Bianchi, Marco Zecca, Luciana Vinti, Elena Facchini, Tommaso Casini, Sayla Bernasconi, Loredana Amoroso, Salvatore D’Amico, Massimo Provenzi, Raffaela De Santis, Antonella Sau, Paola Muggeo, Rosa Maria Mura, Riccardo Haupt, Maurizio Mascarin and Roberta Burnelliadd Show full author list remove Hide full author list
Cancers 2022, 14(6), 1471; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14061471 - 13 Mar 2022
Cited by 3 | Viewed by 2191
Abstract
The objective of this study was to identify prognostic factors for children and adolescents with relapsed or progressive classical Hodgkin’s lymphoma (cHL) to design salvage therapy tailored to them. We analyzed a homogeneous pediatric population, diagnosed with progressive/relapsed cHL previously enrolled in two [...] Read more.
The objective of this study was to identify prognostic factors for children and adolescents with relapsed or progressive classical Hodgkin’s lymphoma (cHL) to design salvage therapy tailored to them. We analyzed a homogeneous pediatric population, diagnosed with progressive/relapsed cHL previously enrolled in two subsequent protocols of the Italian Association of Pediatric Hematology and Oncology in the period 1996–2016. There were 272 eligible patients, 17.5% of treated patients with cHL. Overall survival (OS) and event-free survival (EFS) after a 10-year follow-up were 65.3% and 53.3%, respectively. Patients with progressive disease (PD), advanced stage at recurrence, and ≥5 involved sites showed a significantly worse OS. PD, advanced stage, and extra-nodal involvement at recurrence were significantly associated with a poorer EFS. Multivariable analysis identified three categories for OS based on the type of recurrence and number of localizations: PD and ≥5 sites: OS 34%; PD and <5 sites: OS 56.5%; relapses: OS 73.6%. Four categories were obtained for EFS based on the type of recurrence and stage: PD and stage 3–4: EFS 25.5%; PD and stage 1–2: EFS 43%; relapse and stage 3–4: EFS 55.4%; relapse and stage 1–2: EFS 72.1%. Patients with PD, in advanced stage, or with ≥5 involved sites had a very poor survival and they should be considered refractory to first- and second-line standard chemotherapy. Probably, they should be considered for more innovative approaches since the first progression. Conversely, patients who relapsed later with localized disease had a better prognosis, and they could be considered for a conservative approach. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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14 pages, 650 KiB  
Article
Prevalence of Sleep Disorders, Risk Factors and Sleep Treatment Needs of Adolescents and Young Adult Childhood Cancer Patients in Follow-Up after Treatment
by Shosha H. M. Peersmann, Martha A. Grootenhuis, Annemieke van Straten, Gerard A. Kerkhof, Wim J. E. Tissing, Floor Abbink, Andrica C. H. de Vries, Jacqueline Loonen, Leontien C. M. Kremer, Gertjan J. L. Kaspers and Raphaële R. L. van Litsenburg
Cancers 2022, 14(4), 926; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14040926 - 13 Feb 2022
Cited by 4 | Viewed by 2983
Abstract
Background: Sleep disorders negatively impact adolescent and young adult childhood cancer patients’ physical and psychosocial health. Early recognition improves timely treatment. We therefore studied the prevalence of subjective sleep disorders, risk factors and sleep treatment needs after completion of childhood cancer treatment. Methods: [...] Read more.
Background: Sleep disorders negatively impact adolescent and young adult childhood cancer patients’ physical and psychosocial health. Early recognition improves timely treatment. We therefore studied the prevalence of subjective sleep disorders, risk factors and sleep treatment needs after completion of childhood cancer treatment. Methods: Childhood cancer patients (12–26 years old), ≥6 months after treatment, were invited to fill out the Holland Sleep Disorders Questionnaire, which distinguishes six sleep disorders in substantial agreement with the International Classification of Sleep Disorders, second edition (ICSD-2). They additionally indicated sleep treatment needs. Prevalence rates and needs were displayed in percentages. Logistic regression models were used for risk factors. Results: 576 patients participated (response rate 55.8%)—49.5% females, mean age 17.0 years, 44.4% hemato-oncology, 31.9% solid tumors, 23.6% neuro-oncology. Prevalence rates were: insomnia (9.6%), circadian rhythm sleep disorder (CRSD; 8.1%), restless legs syndrome (7.6%), parasomnia (3.5%), hypersomnia (3.5%) and sleep-related breathing disorders (1.8%). Female sex, comorbid health conditions and young adulthood seem to be risk factors for sleep disorders, but cancer-related factors were not. Differing per sleep disorder, 42–72% wanted help, but only 0–5.6% received sleep treatment. Conclusions: Insomnia and CRSD were most prevalent. An unmet need for sleep treatment was reported by childhood cancer patients during follow-up. Screening for sleep disorders after cancer might improve access to treatment and patient wellbeing. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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13 pages, 301 KiB  
Article
Sociodemographic and Medical Determinants of Quality of Life in Long-Term Childhood Acute Lymphoblastic Leukemia Survivors Enrolled in EORTC CLG Studies
by Charlotte Sleurs, Jammbe Musoro, Ali Rowsell, Michal Kicinski, Stefan Suciu, Sofia Chantziara, Corneel Coens, Madeline Pe, Pierre Missotten, Els Vandecruys, Anne Uyttebroeck, Marie-Françoise Dresse, Claire Pluchart, Alina Ferster, Claire Freycon, Jutte van der Werff ten Bosch, Pierre-Simon Rohrlich, Yves Benoit, Anne-Sophie Darlington and Caroline Piette
Cancers 2022, 14(1), 152; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14010152 - 29 Dec 2021
Cited by 8 | Viewed by 2821
Abstract
Background: due to increasing survival rates in childhood acute lymphoblastic leukemia (ALL), the number of survivors has been expanding. A significant proportion of these survivors can experience long-term emotional and psychosocial problems. However, the exact risk factors remain inconclusive. We investigated potential risk [...] Read more.
Background: due to increasing survival rates in childhood acute lymphoblastic leukemia (ALL), the number of survivors has been expanding. A significant proportion of these survivors can experience long-term emotional and psychosocial problems. However, the exact risk factors remain inconclusive. We investigated potential risk factors for decreased daily life quality and life challenges in long-term childhood ALL survivors enrolled between 1971 and 1998 in EORTC studies. Methods: self-report questionnaires were collected from 186 survivors (109 females; mean age at diagnosis 5.62 years, range 0.2–14.7; median time since diagnosis of 20.5 years (12.9–41.6)), including the Short-Form Health Survey (SF-12) and Impact of Cancer-Childhood Survivors (IOC-CS). Multivariable linear regression models were used to assess the impact of gender, age at diagnosis, relapse/second neoplasm, National Cancer Institute (NCI) risk group and cranial radiotherapy on 2 subscales of the SF-12 (physical and mental health) and five subscales of the IOC-CS (life challenges, body and health, personal growth, thinking and memory problems and socializing). Results: mental component scores of SF-12 were not significantly associated with any risk factor. Physical component scores were lower in relapsed, irradiated and NCI high-risk patients. Regarding IOC-CS negative impact subscales, life challenges was more negatively impacted by cancer in female, younger (i.e., <6 years) and relapsed patients. Regarding the positive impact scales, personal growth was more positively impacted in relapsed patients, whereas body and health, and socializing, were less positively impacted in these patients, compared to non-relapsed patients. Socializing was more positively impacted in older patients (>6 years). Conclusions: this study demonstrates that long-term outcomes can be both adverse and positive, depending on the patient’s demographic and clinical characteristics. Younger, female, and relapsed patients might encounter more life challenges years after their disease, while physical challenges could occur more often in relapsed and high-risk patients. Finally, the positive effect on socializing in the older patients sheds new light on the importance of peer interactions for this subgroup. Specific individual challenges thus need specialized support for specific subgroups. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
10 pages, 641 KiB  
Article
Maternal Thyroid Disease and the Risk of Childhood Cancer in the Offspring
by Laura K. Seppälä, Laura-Maria Madanat-Harjuoja, Maarit K. Leinonen, Mitja Lääperi and Kim Vettenranta
Cancers 2021, 13(21), 5409; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers13215409 - 28 Oct 2021
Cited by 5 | Viewed by 2146
Abstract
Maternal thyroid disease, especially hypothyroidism, affects pregnancy and its outcome. In-utero exposure to autoimmune thyroid disease has been reported to associate with childhood ALL in the offspring. We evaluated the risk of childhood cancer in the offspring following exposure to maternal thyroid disease [...] Read more.
Maternal thyroid disease, especially hypothyroidism, affects pregnancy and its outcome. In-utero exposure to autoimmune thyroid disease has been reported to associate with childhood ALL in the offspring. We evaluated the risk of childhood cancer in the offspring following exposure to maternal thyroid disease in a case-control setting using registry data. All patients with their first cancer diagnosis below the age of 20 years were identified from the Finnish Cancer Registry (n = 2037) and matched for sex and birth year at a 1:5 ratio to population controls identified from the Medical Birth Registry (n = 10,185). We collected national information on maternal thyroid disease from the Medical Birth Registry, Care Register for Health Care, Register for Reimbursed Drug Purchases and Register of Special Reimbursements. We used conditional logistic regression to analyze childhood cancer risk in the offspring. The adjusted OR for any childhood cancer was 1.41 (95%, CI 1.00–2.00) comparing the offspring of mothers with hypothyroidism and those with normal thyroid function. The risk of lymphomas was increased (adjusted OR for maternal hypothyroidism 3.66, 95%, CI 1.29–10.38). The results remained stable when mothers with cancer history were excluded from the analyses. Maternal hypothyroidism appears to be associated with an increased risk for childhood lymphoma in the offspring. The association exists even after excluding possible familial cancers. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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14 pages, 2063 KiB  
Article
Opposite Incidence Trends for Differentiated and Medullary Thyroid Cancer in Young Dutch Patients over a 30-Year Time Span
by Chantal A. Lebbink, Medard F. M. van den Broek, Annemiek B. G. Kwast, Joep P. M. Derikx, Miranda P. Dierselhuis, Schelto Kruijff, Thera P. Links, A. S. Paul van Trotsenburg, Gerlof D. Valk, Menno R. Vriens, Annemarie A. Verrijn Stuart, Hanneke M. van Santen and Henrike E. Karim-Kos
Cancers 2021, 13(20), 5104; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers13205104 - 12 Oct 2021
Cited by 9 | Viewed by 1821 | Correction
Abstract
Thyroid cancer is the most common endocrine malignancy in children. A rising incidence has been reported worldwide. Possible explanations include the increased use of enhanced imaging (leading to incidentalomas) and an increased prevalence of risk factors. We aimed to evaluate the incidence and [...] Read more.
Thyroid cancer is the most common endocrine malignancy in children. A rising incidence has been reported worldwide. Possible explanations include the increased use of enhanced imaging (leading to incidentalomas) and an increased prevalence of risk factors. We aimed to evaluate the incidence and survival trends of thyroid cancer in Dutch children, adolescents, and young adults (0–24 years) between 1990 and 2019. The age-standardized incidence rates of differentiated thyroid cancer (DTC, including papillary and follicular thyroid cancer (PTC and FTC, respectively)) and medullary thyroid cancer (MTC), the average annual percentage changes (AAPC) in incidence rates, and 10-year overall survival (OS) were calculated based on data obtained from the nationwide cancer registry (Netherlands Cancer Registry). A total of 839 patients aged 0–24 years had been diagnosed with thyroid carcinoma (PTC: 594 (71%), FTC: 128 (15%), MTC: 114 (14%)) between 1990 and 2019. The incidence of PTC increased significantly over time (AAPC +3.6%; 95%CI +2.3 to +4.8), the incidence rate of FTC showed a stable trend ((AAPC −1.1%; 95%CI −3.4 to +1.1), while the incidence of MTC decreased significantly (AAPC: −4.4% (95%CI −7.3 to −1.5). The 10-year OS was 99.5% (1990–1999) and 98.6% (2000–2009) in patients with DTC and 92.4% (1990–1999) and 96.0% (2000–2009) in patients with MTC. In this nationwide study, a rising incidence of PTC and decreasing incidence of MTC were observed. For both groups, in spite of the high proportion of patients with lymph node involvement at diagnosis for DTC and the limited treatment options for MTC, 10-year OS was high. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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11 pages, 1971 KiB  
Article
Postgraduate Interprofessional Case-Based Learning in Childhood Cancer: A Feasibility Study
by Martha Krogh Topperzer, Marianne Hoffmann, Hanne Bækgaard Larsen, Susanne Rosthøj, Martin Kaj Fridh, Louise Ingerslev Roug, Liv Andres-Jensen, Peter Erik Lokto Pontoppidan, Kjeld Schmiegelow and Jette Led Sørensen
Cancers 2021, 13(17), 4314; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers13174314 - 26 Aug 2021
Cited by 2 | Viewed by 1950
Abstract
This paper presents a feasibility study assessing the acceptability, demand, implementation, and practicality of postgraduate interprofessional case-based learning in childhood cancer at Copenhagen University Hospital—Rigshospitalet. Healthcare professionals included nurses, doctors, social workers, physiotherapists, occupational therapists, pharmacists, pharmacologists, dieticians, nursing assistants, and professionals with [...] Read more.
This paper presents a feasibility study assessing the acceptability, demand, implementation, and practicality of postgraduate interprofessional case-based learning in childhood cancer at Copenhagen University Hospital—Rigshospitalet. Healthcare professionals included nurses, doctors, social workers, physiotherapists, occupational therapists, pharmacists, pharmacologists, dieticians, nursing assistants, and professionals with a supportive function (teachers, secretaries, priests, and daycare workers). All participated in a postgraduate interprofessional case-based learning session. Feasibility was assessed using Bowen’s focus areas of acceptability, demand, implementation, and practicality. Before and after the intervention session, three measurement tools were used 2–3 weeks before participation and 3–4 weeks after participation to collect data: Assessment of Interprofessional Team Collaboration Scale, Readiness for Interprofessional Learning Scale, and Safety Attitudes Questionnaire. Representing 13 occupational groups, 49 participants completed the case-based learning sessions, indicating acceptability and practicality. The pre- and post-intervention questionnaires were completed by 79% of the participants, 88% of whom rated the professional content as good or very good. A change over time was detected on all three scales measuring mean difference post-intervention scores. The outcome measures can be used to assess the effect of the intervention. Postgraduate interprofessional case-based learning in childhood cancer is feasible in terms of acceptability, demand, implementation, and practicality. Implementation requires leadership commitment at all levels. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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Review

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18 pages, 1688 KiB  
Review
Optimizing Rhabdomyosarcoma Treatment in Adolescents and Young Adults
by Atsushi Makimoto
Cancers 2022, 14(9), 2270; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14092270 - 02 May 2022
Cited by 7 | Viewed by 5496
Abstract
Rhabdomyosarcoma (RMS) is the most common form of soft tissue sarcoma in children, but can also develop in adolescents and young adults (AYA). The mainstay of treatment is multi-agent chemotherapy, ideally with concomitant local treatment, including surgical resection and/or radiation therapy. Although most [...] Read more.
Rhabdomyosarcoma (RMS) is the most common form of soft tissue sarcoma in children, but can also develop in adolescents and young adults (AYA). The mainstay of treatment is multi-agent chemotherapy, ideally with concomitant local treatment, including surgical resection and/or radiation therapy. Although most treatment decisions for RMS in AYA are based on scientific evidence accumulated through clinical studies of pediatric RMS, treatment outcomes are significantly inferior in AYA patients than in children. Factors responsible for the significantly poor outcomes in AYA are tumor biology, the physiology specific to the age group concerned, refractoriness to multimodal treatments, and various psychosocial and medical care issues. The present review aims to examine the various issues involved in the treatment and care of AYA patients with RMS, discuss possible solutions, and provide an overview of the literature on the topic with several observations from the author’s own experience. Clinical trials for RMS in AYA are the best way to develop an optimal treatment. However, a well-designed clinical trial requires a great deal of time and resources, especially when targeting such a rare population. Until clinical trials are designed and implemented, and their findings duly analyzed, we must provide the best possible practice for RMS treatment in AYA patients based on our own expertise in manipulating the dosage schedules of various chemotherapeutic agents and administering local treatments in a manner appropriate for each patient. Precision medicine based on state-of-the-art cancer genomics will also form an integral part of this personalized approach. In the current situation, the only way to realize such a holistic treatment approach is to integrate new developments and findings, such as gene-based diagnostics and treatments, with older, fundamental evidence that can be selectively applied to individual cases. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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25 pages, 571 KiB  
Review
Environmental Risk Factors for Childhood Acute Lymphoblastic Leukemia: An Umbrella Review
by Felix M. Onyije, Ann Olsson, Dan Baaken, Friederike Erdmann, Martin Stanulla, Daniel Wollschläger and Joachim Schüz
Cancers 2022, 14(2), 382; https://0-doi-org.brum.beds.ac.uk/10.3390/cancers14020382 - 13 Jan 2022
Cited by 24 | Viewed by 8808
Abstract
Leukemia is the most common type of cancer among children and adolescents worldwide. The aim of this umbrella review was (1) to provide a synthesis of the environmental risk factors for the onset of childhood acute lymphoblastic leukemia (ALL) by exposure window, (2) [...] Read more.
Leukemia is the most common type of cancer among children and adolescents worldwide. The aim of this umbrella review was (1) to provide a synthesis of the environmental risk factors for the onset of childhood acute lymphoblastic leukemia (ALL) by exposure window, (2) evaluate their strength of evidence and magnitude of risk, and as an example (3) estimate the prevalence in the German population, which determines the relevance at the population level. Relevant systematic reviews and pooled analyses were identified and retrieved through PubMed, Web of Science databases and lists of references. Only two risk factors (low doses of ionizing radiation in early childhood and general pesticide exposure during maternal preconception/pregnancy) were convincingly associated with childhood ALL. Other risk factors including extremely low frequency electromagnetic field (ELF-MF), living in proximity to nuclear facilities, petroleum, benzene, solvent, and domestic paint exposure during early childhood, all showed some level of evidence of association. Maternal consumption of coffee (high consumption/>2 cups/day) and cola (high consumption) during pregnancy, paternal smoking during the pregnancy of the index child, maternal intake of fertility treatment, high birth weight (≥4000 g) and caesarean delivery were also found to have some level of evidence of association. Maternal folic acid and vitamins intake, breastfeeding (≥6 months) and day-care attendance, were inversely associated with childhood ALL with some evidence. The results of this umbrella review should be interpreted with caution; as the evidence stems almost exclusively from case-control studies, where selection and recall bias are potential concerns, and whether the empirically observed association reflect causal relationships remains an open question. Hence, improved exposure assessment methods including accurate and reliable measurement, probing questions and better interview techniques are required to establish causative risk factors of childhood leukemia, which is needed for the ultimate goal of primary prevention. Full article
(This article belongs to the Special Issue Childhood and Adolescent Cancer)
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