A 5‐year‐old female diagnosed with severe hemophilia B began experiencing frequent muscular and joint bleeds at 19 months old. Molecular studies, including Sanger sequencing, Giemsa banding, human androgen receptor (HUMARA) assay, array‐based comparative genomic hybridization (aCGH), whole‐exome sequencing (WES), and multiplex ligation‐dependent probe amplification (MLPA), revealed a heterozygous factor IX (F9) intron 3 substitution (c.277+1G>T) inherited from her mother and a de novo heterozygous 441 kb deletion in the Xq28 region, which flanked intron 22 homologous regions 1 (int22h1) and 2 (int22h2). This rare genetic profile explains her severe phenotype and guides hereditary consultation for family planning.
{"title":"A de novo int22h‐1/int22h‐2‐flanked Xq28 deletion‐associated preferential X‐inactivation in a female with severe hemophilia B","authors":"Wan‐Chun Chen, Hsiao‐Jung Kao, Pui‐Yan Kwok, Shyh‐Shin Chiou, Yu‐Ling Kuo, Wan‐Yi Hsu, Ping‐Tao Lu, Cian‐Rong Wu, Pei‐Chin Lin","doi":"10.1002/pbc.31332","DOIUrl":"https://doi.org/10.1002/pbc.31332","url":null,"abstract":"A 5‐year‐old female diagnosed with severe hemophilia B began experiencing frequent muscular and joint bleeds at 19 months old. Molecular studies, including Sanger sequencing, Giemsa banding, human androgen receptor (HUMARA) assay, array‐based comparative genomic hybridization (aCGH), whole‐exome sequencing (WES), and multiplex ligation‐dependent probe amplification (MLPA), revealed a heterozygous factor IX (F9) intron 3 substitution (c.277+1G>T) inherited from her mother and a de novo heterozygous 441 kb deletion in the Xq28 region, which flanked intron 22 homologous regions 1 (int22h1) and 2 (int22h2). This rare genetic profile explains her severe phenotype and guides hereditary consultation for family planning.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259660","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ahmed Farrag, Mohammad R. Alqudimat, Fatma A. A. Hassan
Background and purposeThe use of traditional and complementary medicine (T&CM) is common in children with cancer globally. We aimed to assess the prevalence, types, reasons, perceived effectiveness, and disclosure rate of T&CM use among children with cancer in Southern Egypt. We also investigated whether T&CM use contributed to delays in initial presentation and treatment.Materials and methodsA cross‐sectional design was utilized. Data were collected via an interviewer‐administered questionnaire. Eligible children and their caregivers at the South Egypt Cancer Institute were invited to participate.ResultsEighty‐six children completed the study (response rate = 86%). T&CM use was reported by 52 (60.5%) patients, with six (11.5%) experienced delayed presentation. The reasons for T&CM use were complementary for 37 (71%) and alternative for 15 (29%) of the participants. The types of T&CM used included herbal (63%), nutritional (33%), witchcraft (29%), and religious (19%) therapies. Approximately 48% of users employed multiple T&CM types. Family members recommended T&CM for 60% of the users. Most patients (65%) perceived T&CM as effective, with 71% initiating its use during the early phases of treatment. Almost all participants (98%) reported that healthcare providers did not inquire about T&CM use. T&CM usage was more prevalent among wealthier families (p = .023). There was no significant relationship between T&CM use and patient gender, diagnosis, residence, or paternal educational level.ConclusionsThe significant utilization of T&CM among children with cancer highlights the need for healthcare providers to engage in open and early discussions with families regarding T&CM use.
{"title":"Family strategies for managing childhood cancer: Using traditional and complementary medicine in Southern Egypt","authors":"Ahmed Farrag, Mohammad R. Alqudimat, Fatma A. A. Hassan","doi":"10.1002/pbc.31326","DOIUrl":"https://doi.org/10.1002/pbc.31326","url":null,"abstract":"Background and purposeThe use of traditional and complementary medicine (T&CM) is common in children with cancer globally. We aimed to assess the prevalence, types, reasons, perceived effectiveness, and disclosure rate of T&CM use among children with cancer in Southern Egypt. We also investigated whether T&CM use contributed to delays in initial presentation and treatment.Materials and methodsA cross‐sectional design was utilized. Data were collected via an interviewer‐administered questionnaire. Eligible children and their caregivers at the South Egypt Cancer Institute were invited to participate.ResultsEighty‐six children completed the study (response rate = 86%). T&CM use was reported by 52 (60.5%) patients, with six (11.5%) experienced delayed presentation. The reasons for T&CM use were complementary for 37 (71%) and alternative for 15 (29%) of the participants. The types of T&CM used included herbal (63%), nutritional (33%), witchcraft (29%), and religious (19%) therapies. Approximately 48% of users employed multiple T&CM types. Family members recommended T&CM for 60% of the users. Most patients (65%) perceived T&CM as effective, with 71% initiating its use during the early phases of treatment. Almost all participants (98%) reported that healthcare providers did not inquire about T&CM use. T&CM usage was more prevalent among wealthier families (<jats:italic>p</jats:italic> = .023). There was no significant relationship between T&CM use and patient gender, diagnosis, residence, or paternal educational level.ConclusionsThe significant utilization of T&CM among children with cancer highlights the need for healthcare providers to engage in open and early discussions with families regarding T&CM use.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259666","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Megan M. Lilley, Marta Salek, Ashley Holland, Hiroto Inaba, Sara M. Federico
{"title":"Treatment of a pediatric patient with concurrent neuroblastoma and acute lymphoblastic leukemia","authors":"Megan M. Lilley, Marta Salek, Ashley Holland, Hiroto Inaba, Sara M. Federico","doi":"10.1002/pbc.31313","DOIUrl":"https://doi.org/10.1002/pbc.31313","url":null,"abstract":"","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259665","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Incidental diagnosis of pediatric chronic myeloid leukemia in the setting of acute appendicitis: A case report and review of management strategies","authors":"Zhidong Wang, Jake Gigliotti, Christine Gough, Claire Gallion, Afshin Ameri","doi":"10.1002/pbc.31322","DOIUrl":"https://doi.org/10.1002/pbc.31322","url":null,"abstract":"","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259661","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
BackgroundVeno‐occlusive disease (VOD), also known as sinusoidal obstruction syndrome (SOS), is a life‐threatening complications of hematopoietic cell transplantation (HCT).MethodsWe studied the impact of early defibrotide (DF) therapy on the outcomes of pediatric patients with VOD/SOS after transplantation, focusing on recent immunotherapies. A total of 111 pediatric patients who underwent HCT for malignant disease between February 2017 and March 2023 at Kyushu University Hospital were included.ResultsAmong 111 patients of less than 20 years of age who underwent HCT for malignancy at a single institution between 2017 and 2023, VOD/SOS occurred in 25 (23%) patients. VOD/SOS developed more frequently in the post‐DF era (2020–2023, n = 58) than in the pre‐DF era (31% vs. 13%, p = .04). The proportion of patients with relapsed/refractory acute lymphoblastic leukemia (ALL) was higher in the post‐DF era than in the pre‐DF era (44% vs. 8%, p = .04). Early DF therapy that was started at two European Society for Blood and Marrow Transplantation diagnostic criteria reduced the severity of VOD/SOS (p < .01) in comparison to non‐early therapy started at less than two criteria. A multivariate analysis indicated that a history of cytokine release syndrome (odds ratio [OR] = 10.4, p = .01) and juvenile myelomonocytic leukemia (OR = 8.98, p = .04), but not an endothelial activation and stress index (EASIX) score of greater than 0.85, were independent risk factors for VOD/SOS.ConclusionsEarly DF therapy improves the severity and survival outcomes of post‐transplant VOD/SOS in children. However, its incidence is increasing in the era of immunotherapy for progressive diseases.
{"title":"Early defibrotide therapy and risk factors for post‐transplant veno‐occlusive disease/sinusoidal obstruction syndrome in childhood","authors":"Hironori Goto, Utako Oba, Tamaki Ueda, Shunsuke Yamamoto, Masataka Inoue, Yu Shimo, Satomi Yokoyama, Yusuke Takase, Wakako Kato, Souichi Suenobu, Kenji Ihara, Yuhki Koga, Shouichi Ohga","doi":"10.1002/pbc.31331","DOIUrl":"https://doi.org/10.1002/pbc.31331","url":null,"abstract":"BackgroundVeno‐occlusive disease (VOD), also known as sinusoidal obstruction syndrome (SOS), is a life‐threatening complications of hematopoietic cell transplantation (HCT).MethodsWe studied the impact of early defibrotide (DF) therapy on the outcomes of pediatric patients with VOD/SOS after transplantation, focusing on recent immunotherapies. A total of 111 pediatric patients who underwent HCT for malignant disease between February 2017 and March 2023 at Kyushu University Hospital were included.ResultsAmong 111 patients of less than 20 years of age who underwent HCT for malignancy at a single institution between 2017 and 2023, VOD/SOS occurred in 25 (23%) patients. VOD/SOS developed more frequently in the post‐DF era (2020–2023, <jats:italic>n</jats:italic> = 58) than in the pre‐DF era (31% vs. 13%, <jats:italic>p </jats:italic>= .04). The proportion of patients with relapsed/refractory acute lymphoblastic leukemia (ALL) was higher in the post‐DF era than in the pre‐DF era (44% vs. 8%, <jats:italic>p </jats:italic>= .04). Early DF therapy that was started at two European Society for Blood and Marrow Transplantation diagnostic criteria reduced the severity of VOD/SOS (<jats:italic>p </jats:italic>< .01) in comparison to non‐early therapy started at less than two criteria. A multivariate analysis indicated that a history of cytokine release syndrome (odds ratio [OR] = 10.4, <jats:italic>p </jats:italic>= .01) and juvenile myelomonocytic leukemia (OR = 8.98, <jats:italic>p </jats:italic>= .04), but not an endothelial activation and stress index (EASIX) score of greater than 0.85, were independent risk factors for VOD/SOS.ConclusionsEarly DF therapy improves the severity and survival outcomes of post‐transplant VOD/SOS in children. However, its incidence is increasing in the era of immunotherapy for progressive diseases.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259662","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Joel Milam, Yoonji Kim, Michael Roth, David R. Freyer
Lifelong, guideline‐based monitoring for late effects is recommended for childhood cancer survivors (CCS). We examined rates of receiving surveillance tests among at‐risk young adult CCS in a population‐based study (n = 253; 50% Hispanic/Latino; mean post‐treatment interval 14.5 years, range: 5–22). Adherence rates were 36.1%, 31.9%, and 36.4% among those indicated for cardiac (n = 119), thyroid (n = 68), and breast (n = 66) surveillance, respectively, indicating that poor surveillance among long‐term CCS is widespread. Receipt of any of these surveillance tests was positively associated with being in follow‐up care, having any health insurance (vs. none), and receiving education about need for follow‐up with surveillance (all p‐values less than .05).
{"title":"Late effects surveillance adherence among young adult childhood cancer survivors: A population‐based study","authors":"Joel Milam, Yoonji Kim, Michael Roth, David R. Freyer","doi":"10.1002/pbc.31328","DOIUrl":"https://doi.org/10.1002/pbc.31328","url":null,"abstract":"Lifelong, guideline‐based monitoring for late effects is recommended for childhood cancer survivors (CCS). We examined rates of receiving surveillance tests among at‐risk young adult CCS in a population‐based study (<jats:italic>n</jats:italic> = 253; 50% Hispanic/Latino; mean post‐treatment interval 14.5 years, range: 5–22). Adherence rates were 36.1%, 31.9%, and 36.4% among those indicated for cardiac (<jats:italic>n</jats:italic> = 119), thyroid (<jats:italic>n</jats:italic> = 68), and breast (<jats:italic>n</jats:italic> = 66) surveillance, respectively, indicating that poor surveillance among long‐term CCS is widespread. Receipt of any of these surveillance tests was positively associated with being in follow‐up care, having any health insurance (vs. none), and receiving education about need for follow‐up with surveillance (all <jats:italic>p</jats:italic>‐values less than .05).","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259663","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Wajiha J. Kheir, Andre Slim, Dalia El Hadi, Elie Bechara, Raphah Borghol, Dolly Noun, Christiane Al‐Haddad
BackgroundRetinoblastoma, the most common intraocular malignancy in children, has high fatality rates if untreated. It is crucial to monitor treatment effectiveness and explore factors influencing favorable outcomes. Our study aims to examine how tumor location impacts the response to standard treatments and the achievement of favorable outcomes among retinoblastoma patients, while controlling for other tumor‐related factors.MethodsThis retrospective study analyzed medical records of retinoblastoma patients from November 2012 to December 2022 enrolled in the retinoblastoma program at the Children's Cancer Center of Lebanon (established in collaboration with St.y Jude Children's Research Hospital, Memphis, TN). Data were extracted from the electronic chart reviews and operative reports of examinations under anesthesia (EUAs), and included patient's demographics, tumor characteristics (size, location), and treatment parameters (treatment type, resolution, recurrence).ResultsThe study included 42 patients with retinoblastoma, with a total of 57 eyes and 115 tumors/lesions. The median age at diagnosis was 12 months (range: 2–36 months). Among the patients, 26 (61.9%) were males and 16 (38.1%) were females. A minority of patients (21.4%) presented with unilateral involvement, whereas the majority (78.6%) had bilateral involvement. The locations of retinoblastoma lesions were distributed as follows: optic nerve (4.4%), macula (19.1%), superior (16.5%), inferior (17.4%), nasal (27.8%), and temporal (14.8%). Resolution rate tended to be highest for tumors close to optic nerve and temporal lesions, but no statistical significance was attained (p = .45). Macular lesions tended to have the fastest resolution, but again not significantly (p = .5). Multiple logistic regression revealed that the odds for resolution of tumor was not significantly associated with tumor size (p = .57) or location (p = .52).ConclusionLocation of retinoblastoma lesions was not directly associated with recurrence‐free resolution in our cohort. Further research in large retinoblastoma databases is needed to explore the association of tumor characteristics with recurrence and the need for secondary therapeutic interventions.
{"title":"Treatment outcomes in retinoblastoma and the effect of tumor topography","authors":"Wajiha J. Kheir, Andre Slim, Dalia El Hadi, Elie Bechara, Raphah Borghol, Dolly Noun, Christiane Al‐Haddad","doi":"10.1002/pbc.31334","DOIUrl":"https://doi.org/10.1002/pbc.31334","url":null,"abstract":"BackgroundRetinoblastoma, the most common intraocular malignancy in children, has high fatality rates if untreated. It is crucial to monitor treatment effectiveness and explore factors influencing favorable outcomes. Our study aims to examine how tumor location impacts the response to standard treatments and the achievement of favorable outcomes among retinoblastoma patients, while controlling for other tumor‐related factors.MethodsThis retrospective study analyzed medical records of retinoblastoma patients from November 2012 to December 2022 enrolled in the retinoblastoma program at the Children's Cancer Center of Lebanon (established in collaboration with St.y Jude Children's Research Hospital, Memphis, TN). Data were extracted from the electronic chart reviews and operative reports of examinations under anesthesia (EUAs), and included patient's demographics, tumor characteristics (size, location), and treatment parameters (treatment type, resolution, recurrence).ResultsThe study included 42 patients with retinoblastoma, with a total of 57 eyes and 115 tumors/lesions. The median age at diagnosis was 12 months (range: 2–36 months). Among the patients, 26 (61.9%) were males and 16 (38.1%) were females. A minority of patients (21.4%) presented with unilateral involvement, whereas the majority (78.6%) had bilateral involvement. The locations of retinoblastoma lesions were distributed as follows: optic nerve (4.4%), macula (19.1%), superior (16.5%), inferior (17.4%), nasal (27.8%), and temporal (14.8%). Resolution rate tended to be highest for tumors close to optic nerve and temporal lesions, but no statistical significance was attained (<jats:italic>p</jats:italic> = .45). Macular lesions tended to have the fastest resolution, but again not significantly (<jats:italic>p</jats:italic> = .5). Multiple logistic regression revealed that the odds for resolution of tumor was not significantly associated with tumor size (<jats:italic>p</jats:italic> = .57) or location (<jats:italic>p</jats:italic> = .52).ConclusionLocation of retinoblastoma lesions was not directly associated with recurrence‐free resolution in our cohort. Further research in large retinoblastoma databases is needed to explore the association of tumor characteristics with recurrence and the need for secondary therapeutic interventions.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259673","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Jane Koo, John Huber, Priscila Badia, Chloe Dunseath, Gabby O'Connor, Stella M. Davies, Christopher E. Dandoy
BackgroundPediatric and young adult patients undergoing autologous hematopoietic stem cell transplant (auto‐HSCT) face a crucial, yet understudied, risk of invasive fungal infections (IFI), especially compared to allogeneic transplants. This gap underscores the need for research in pediatric patients undergoing auto‐HSCT. Our objective was to evaluate the incidence of IFI in pediatric and young adult patients during the first year after auto‐HSCT.Materials and methodsWe conducted a single‐center retrospective analysis of 150 pediatric and young adult auto‐HSCT patients who underwent transplant from January 2013 to January 2023. We focused on IFI incidence within the first‐year post transplant, using the European Organization for Research and Treatment of Cancer/Mycoses Study Group (EORTC/MSG) criteria for IFI identification.ResultsAmong the 150 patients analyzed, with 240 unique transplant episodes, the primary indication was neuroblastoma (37.3%), and micafungin was extensively used for prophylaxis (82.7%). There was an absence of IFI from yeast and mold species, suggesting a low IFI risk in this cohort. The incidence of IFI in pediatric auto‐HSCT recipients receiving micafungin primary antifungal prophylaxis is rare.ConclusionsThe findings advocate for further research to refine prophylaxis guidelines and highlight the need for individualized risk assessment to optimize post‐transplant care.
{"title":"Invasive fungal infections are rare in pediatric and young adult autologous hematopoietic stem cell transplant patients","authors":"Jane Koo, John Huber, Priscila Badia, Chloe Dunseath, Gabby O'Connor, Stella M. Davies, Christopher E. Dandoy","doi":"10.1002/pbc.31336","DOIUrl":"https://doi.org/10.1002/pbc.31336","url":null,"abstract":"BackgroundPediatric and young adult patients undergoing autologous hematopoietic stem cell transplant (auto‐HSCT) face a crucial, yet understudied, risk of invasive fungal infections (IFI), especially compared to allogeneic transplants. This gap underscores the need for research in pediatric patients undergoing auto‐HSCT. Our objective was to evaluate the incidence of IFI in pediatric and young adult patients during the first year after auto‐HSCT.Materials and methodsWe conducted a single‐center retrospective analysis of 150 pediatric and young adult auto‐HSCT patients who underwent transplant from January 2013 to January 2023. We focused on IFI incidence within the first‐year post transplant, using the European Organization for Research and Treatment of Cancer/Mycoses Study Group (EORTC/MSG) criteria for IFI identification.ResultsAmong the 150 patients analyzed, with 240 unique transplant episodes, the primary indication was neuroblastoma (37.3%), and micafungin was extensively used for prophylaxis (82.7%). There was an absence of IFI from yeast and mold species, suggesting a low IFI risk in this cohort. The incidence of IFI in pediatric auto‐HSCT recipients receiving micafungin primary antifungal prophylaxis is rare.ConclusionsThe findings advocate for further research to refine prophylaxis guidelines and highlight the need for individualized risk assessment to optimize post‐transplant care.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142259668","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Danielle Novetsky Friedman, Jessica A. Lavery, Chaya S. Moskowitz, Isabella Gordon, Jaime Gilliland, Jessica Scott, Deborah Diotallevi, Elaine Pottenger, Nadia Wilson, Zoltan Antal, Sameera Ramjan, Stephen Sands
BackgroundLate morbidity after childhood cancer may be mitigated by healthy lifestyle behaviors. We piloted a game‐based, parent–child digital intervention to increase activity in sedentary survivors.MethodsSurvivors aged 10–16 treated with cardiotoxic therapy and not meeting US physical activity guidelines were enrolled in a single‐arm study with a parent. Following a 14‐day run‐in, participants chose a prespecified step goal and wore an accelerometer daily for 24 weeks (12‐week game‐based intervention; 12‐week follow‐up). Participants completed the Acceptability of Intervention Measure (AIM) at 24 weeks; a subset of dyads completed qualitative interviews.ResultsAmong 129 eligible survivors, 27 enrolled (20.9% participation rate) with a parent. Four dyads were removed during the 14‐day run‐in period due to noncompliance. Among the 23 dyads who continued to the study, the tracker was worn for 95% of days during the 12‐week intervention (95% confidence interval [CI]: 94–96) and 81% during the 12‐week follow‐up (95% CI: 79–82). Overall, the prespecified step goal was met for 64% (95% CI: 63–66) of days during the intervention and 37% (95% CI: 35–38) during the follow‐up. At the end of study, 17/23 dyads responded to AIM; 82% of survivors and 94% of parents reported the intervention as acceptable. During qualitative interviews (n = 5), dyads noted that they liked the accountability of the “buddy system,” but would have liked more personalized goal‐setting.ConclusionsDespite high ratings of acceptability among participants, difficulties with sustained adherence and retention were encountered in this parent–child gamification intervention. Alternate, tailored designs should be considered in the future.
{"title":"Feasibility and acceptability of a parent–child intervention to improve step count in childhood cancer survivors exposed to cardiotoxic therapy: The STEP UP for FAMILIES Study","authors":"Danielle Novetsky Friedman, Jessica A. Lavery, Chaya S. Moskowitz, Isabella Gordon, Jaime Gilliland, Jessica Scott, Deborah Diotallevi, Elaine Pottenger, Nadia Wilson, Zoltan Antal, Sameera Ramjan, Stephen Sands","doi":"10.1002/pbc.31320","DOIUrl":"https://doi.org/10.1002/pbc.31320","url":null,"abstract":"BackgroundLate morbidity after childhood cancer may be mitigated by healthy lifestyle behaviors. We piloted a game‐based, parent–child digital intervention to increase activity in sedentary survivors.MethodsSurvivors aged 10–16 treated with cardiotoxic therapy and not meeting US physical activity guidelines were enrolled in a single‐arm study with a parent. Following a 14‐day run‐in, participants chose a prespecified step goal and wore an accelerometer daily for 24 weeks (12‐week game‐based intervention; 12‐week follow‐up). Participants completed the Acceptability of Intervention Measure (AIM) at 24 weeks; a subset of dyads completed qualitative interviews.ResultsAmong 129 eligible survivors, 27 enrolled (20.9% participation rate) with a parent. Four dyads were removed during the 14‐day run‐in period due to noncompliance. Among the 23 dyads who continued to the study, the tracker was worn for 95% of days during the 12‐week intervention (95% confidence interval [CI]: 94–96) and 81% during the 12‐week follow‐up (95% CI: 79–82). Overall, the prespecified step goal was met for 64% (95% CI: 63–66) of days during the intervention and 37% (95% CI: 35–38) during the follow‐up. At the end of study, 17/23 dyads responded to AIM; 82% of survivors and 94% of parents reported the intervention as acceptable. During qualitative interviews (<jats:italic>n</jats:italic> = 5), dyads noted that they liked the accountability of the “buddy system,” but would have liked more personalized goal‐setting.ConclusionsDespite high ratings of acceptability among participants, difficulties with sustained adherence and retention were encountered in this parent–child gamification intervention. Alternate, tailored designs should be considered in the future.","PeriodicalId":19822,"journal":{"name":"Pediatric Blood & Cancer","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142269317","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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