Extracranial germ cell tumours in children and adolescents: Results from the French TGM13 protocol

Background

Chemotherapy for non-seminomatous germ cell tumours (NSGCT) exposes to dose-dependent toxicities. The TGM13-NS protocol (EudraCT 2013-004039-60) aimed to decrease the chemotherapy burden compared to the previous TGM95 protocol while maintaining the 5-year event-free survival (EFS) at 80% or more.

Procedure

Patients less than 19 years of age with disseminated NSGCT were enrolled (May 2014 to May 2019) and stratified into four groups: two intermediate-risk (IR: localised tumour with low tumour markers [TM]) groups treated with VBP (vinblastine–bleomycin–cisplatin): three courses for IR1 (ovarian tumour any age/testis tumour less than or equal to 10 years) and four courses for IR2 (extragonadal tumour 10 years or less) groups, and two high-risk (HR: metastatic and/or high TM) groups treated with etoposide–cisplatin and either ifosfamide (VIP) or bleomycin (BEP): three courses for HR1 (ovarian tumour any age/testis tumour less than or equal to 10 years and low TM/testis tumour more than 10 years and very low TM) groups and four courses for HR2 (remainder) groups.

Results

One hundred fifteen patients were included: median age of 12.8 years (0.4–18.9); tumour sites: 44 ovaries, 37 testes and 34 extragonadal. The 5-year EFS and overall survival (OS) were 87% (95% CI: 80–92) and 95% (89–98), respectively (median follow-up: 3.5 years, range: 0.2–5.9), similar to those of the TGM95 protocol (5-year EFS 89% (84–93), 5-year OS 93% (89–95), p = .561). The 5-year EFS were 93% (95% CI: 80–98), 88% (71–95) and 79% (62–90) for ovarian, testicular and extragonadal tumours, respectively. The 5-year EFS varied (p = .02) according to the risk groups: 90% (66–97), 64% (30–85), 95% (72–99) and 87% (74–94) for IR1, IR2, HR1 and HR2, respectively. TM decline adjusted to tumour site, and alpha-fetoprotein (AFP) level revealed a prognostic impact of time to normalisation on EFS: HR = 1.03 (1.003–1.007).

Conclusion

Risk-adapted and globally decreased chemotherapy burden maintains excellent outcomes, exclusive of the IR2 group, which warrants more intensive chemotherapy.     

Worries and anxiety in parents of adult survivors of childhood cancer: A report from the Swiss Childhood Cancer Survivor Study-Parents

Objective

Having a child diagnosed with cancer is distressing for parents. We aimed to compare worries and anxiety in parents of adult childhood cancer survivors with parents of the Swiss general population (GP-parents), and to evaluate characteristics associated with worry in parents of survivors.

Methods

We conducted a nationwide, population-based study in parents of survivors (survivors aged ≥20 years at study, ≤16 years at diagnosis, >5 years post diagnosis) and GP-parents (≥1 child aged ≥20 years at study). We used the Worry and Anxiety Questionnaire (WAQ), and computed the WAQ total score (worries; possible range 0–80) and caseness for generalized anxiety disorder (anxiety), cognitive, somatic, and any criteria. We used multilevel, multivariable linear regression to identify characteristics associated with worries in parents of survivors.

Results

We included 787 parents of 513 survivors (41.0% fathers) and 478 GP-parents (42.3% fathers). Parents of survivors and GP-parents did not differ regarding worries (16.6 vs. 17.1, p = .977), anxiety (2.7% vs. 3.6%, p = .536), cognitive (p = .440), and somatic criteria (p = .067). Less parents of survivors met any criteria (17.7% vs. 24.0%, p = .039). Half of parents reported current cancer-related worries. Higher cancer-related worries were reported by mothers (β = 4.1; 95% CI: 2.0–6.2), parents with one child (β = 5.9; 95% CI: 2.0–9.7), currently experiencing disadvantages because of their child's former disease (β = 7.3; 95% CI: 4.0–10.6), or with support needs (β = 9.0; 95% CI: 3.9–14.2; p = .001).

Conclusions

It is encouraging that most parents of adult survivors report similar worries and anxiety as GP-parents, but cancer-related worries are still prevalent. Efforts should be made to empower parents to seek psycho-social support if required.     

Neurocognitive deficits may not resolve following pediatric kidney transplantation

Background

Pediatric chronic kidney disease (CKD) patients are at risk for cognitive deficits with worsening disease progression. Limited, existing cross-sectional studies suggest that cognitive deficits may improve following kidney transplantation. We sought to assess cognitive performance in relationship to kidney transplantation and kidney-specific medical variables in a sample of pediatric kidney transplant patients who provided cross-sectional and longitudinal observations.

Methods

A retrospective chart review was conducted in patients who completed pre- and/or post-transplant neurocognitive testing at the University of Iowa from 2015–2021. Cognitive outcomes were investigated with developmentally appropriate, standardized measures. Mixed linear models estimated the impact of transplant status on cognitive function (z-scores). Subsequent post-hoc t-tests on change scores were limited to patients who had provided pre- and post-transplant assessments.

Results

Thirty eight patients underwent cognitive assessments: 10 had both pre- and post-transplant cognitive assessments, 11 had pre-transplant assessments only, and 17 had post-transplant data only. Post-transplant status was associated with significantly lower full-scale IQ and slower processing speed compared to pre-transplant status (estimate = −0.32, 95% confidence interval [CI] = −0.52: −0.12; estimate = −0.86, CI = −1.17: −0.55, respectively). Post-hoc analyses confirmed results from the mixed models (FSIQ change score = −0.34, 95% CI = −0.56: −0.12; processing speed change score = −0.98, CI = −1.28: −0.68). Finally, being ≥80 months old at transplant was associated with substantially lower FSIQ compared to being <80 months (estimate = −1.25, 95% CI = −1.94: −0.56).

Conclusions

Our results highlight the importance of monitoring cognitive function following pediatric kidney transplant and identify older transplant age as a risk factor for cognitive deficits.     

Facial deformation following treatment for pediatric head and neck rhabdomyosarcoma; the difference between treatment modalities. Results of a trans-Atlantic,multicenter cross-sectional cohort study

Background

The four different local therapy strategies used for head and neck rhabdomyosarcoma (HNRMS) include proton therapy (PT), photon therapy (RT), surgery with radiotherapy (Paris-method), and surgery with brachytherapy (AMORE). Local control and survival is comparable; however, the impact of these different treatments on facial deformation is still poorly understood. This study aims to quantify facial deformation and investigates the differences in facial deformation between treatment modalities.

Methods

Across four European and North American institutions, HNRMS survivors treated between 1990 and 2017, more than 2 years post treatment, had a 3D photograph taken. Using dense surface modeling, we computed facial signatures for each survivor to show facial deformation relative to 35 age–sex–ethnicity-matched controls. Additionally, we computed individual facial asymmetry.

Findings

A total of 173 HNRMS survivors were included, survivors showed significantly reduced facial growth (p < .001) compared to healthy controls. Partitioned by tumor site, there was reduced facial growth in survivors with nonparameningeal primaries (p = .002), and parameningeal primaries (p ≤.001), but not for orbital primaries (p = .080) All patients were significantly more asymmetric than healthy controls, independent of treatment modality (p ≤ .001). There was significantly more facial deformation in orbital patients when comparing RT to AMORE (p = .046). In survivors with a parameningeal tumor, there was significantly less facial deformation in PT when compared to RT (p = .009) and Paris-method (p = .007).

Interpretation

When selecting optimal treatment, musculoskeletal facial outcomes are an expected difference between treatment options. These anticipated differences are currently based on clinicians’ bias, expertise, and experience. These data supplement clinician judgment with an objective analysis highlighting the impact of patient age and tumor site between existing treatment options.     

Misclassification of self‐reported smoking in adult survivors of childhood cancer

We investigated misclassification rates, sensitivity, and specificity of self‐reported cigarette smoking through serum cotinine concentration (liquid chromatography tandem mass spectrometry) among 287 adult survivors of childhood cancer. Overall, 2.5–6.7% and 19.7–36.9% of the self‐reported never and past smokers had cotinine levels indicative of active smoking. Sensitivity and specificity of self‐reported smoking were 57.5–67.1% and 96.6–99.2%. Misclassification was associated with younger age (OR = 3.2; 95% CI = 1.4–7.4), male (OR = 2.1; 95% CI = 1.1–4.0), and past (OR = 2.7; 95% CI = 1.3–5.8) or current (OR = 2.6; 95% CI = 1.0–6.6) marijuana use. After adjusting for tobacco‐related variables, current marijuana use remained a significant risk for misclassification. Clinicians/researchers should consider bio‐verification to measure smoking status among survivors.     

Danaparoid reduces transplant‐related mortality in stem cell transplantation for children

In SCT, death from transplant‐related complications is the major obstacle hindering improvement of transplant outcomes, and proper supportive care is essential to reduce TRM. The transplant outcomes of 210 pediatric patients with malignant and non‐malignant disorders who consecutively underwent SCT in our institution from 2000 to 2013 were analyzed. The transplant years were divided into three periods: A (2000‐2004), B (2005‐2008), and C (2009‐2013), and an improvement in 5‐year OS and a decrease in 5‐year TRM were observed over these time periods; that is, OS was 61.5%, 60.3%, and 79.5% (P = .062), and TRM was 19.9%, 7.9%, and 0.0% (P < .001) in periods A, B, and C, respectively. On multivariate analysis, the prognostic factor for TRM for all patients was administration of danaparoid (HR = 0.109, 95% CI = 0.033‐0.363, P < .001), and for patients with hematological malignancies in allogeneic SCT, the prognostic factors were danaparoid (HR = 0.046, 95% CI = 0.006‐0.326, P = .002) and advanced disease at SCT (HR = 4.802, 95% CI = 1.734‐13.30, P = .003). A reduction in TRM after SCT was observed over the time periods, and supportive care with danaparoid was found to be significantly effective in reducing TRM in SCT for children.     

The prognostic value of IKZF1plus in B-cell progenitor acute lymphoblastic leukemia: Results from the EORTC 58951 trial

Background

IKZF1 gene deletion is an indicator of poor prognosis in childhood B-cell precursor acute lymphoblastic leukemia (BCP-ALL). The AEIOP/BFM group proposed that the prognostic strength of IKZF1 deletion could be remarkably improved by taking into account additional genetic deletions and reported that among patients with an IKZF1 deletion those with deletions in CDKN2A/2B, PAX5, or PAR1 in the absence of ERG deletion, grouped as IKZF1^plus, had the worst outcome.

Procedure

Between 1998 and 2008, 1636 patients under 18 years of age with previously untreated BCP-ALL were registered in the EORTC 58951 trial. Those with multiplex ligation-dependent probe amplification data were included in this analysis. Unadjusted and adjusted Cox model was used to investigate the additional prognostic value of IKZF1^plus.

Results

Among 1200 patients included in the analysis, 1039 (87%) had no IKZF1 deletion (IKZF1^WT), 87 (7%) had an IKZF1 deletion but not IKZF1^plus (IKZF1^del) and 74 (6%) had IKZF1^plus. In the unadjusted analysis, both patients with IKZF1^del (hazard ratio [HR] = 2.10, 95% confidence interval [CI]: 1.34–3.31) and IKZF1^plus (HR = 3.07, 95% CI: 2.01–4.67) had a shorter event-free survival compared with IKZF1^WT. However, although the IKZF1^plus status was associated with patients’ characteristics indicating poor prognosis, the difference between IKZF1^plus and IKZF1^del was not statistically significant (HR = 1.46, 95% CI: 0.83–2.57, p = .19). The results of the adjusted analysis were similar to the unadjusted analysis.

Conclusions

In patients with BCP-ALL from the EORTC 58951 trial, the improvement of the prognostic importance of IKZF1 by considering IKZF1^plus was not statistically significant.     

Impact of extremely low‐birth‐weight status on risk and resilience for depression and anxiety in adulthood

Background

Preterm birth is associated with an increased risk of depression and anxiety, but it is not known if this is due to greater exposure to risk, or if perinatal adversity amplifies the impact of traditional risk factors. This study sought to determine if exposure to perinatal adversity modifies associations between traditional risk and resilience factors and depression and anxiety in adulthood.

Methods

A sample of 142 extremely low‐birth‐weight (ELBW < 1,000 g) survivors and 133 sociodemographically matched normal birth weight (NBW) control participants was followed longitudinally to 22–26 years of age. Separate postnatal risk and resilience scales were created using eight risk and seven resilience factors, respectively. Depression and anxiety were assessed using the internalizing scale of the Young Adult Self‐Report (YASR). This scale was also dichotomized at the 90th percentile to define clinically significant psychopathology.

Results

While the average number of risk exposures did not differ between groups, ELBW survivors were more susceptible to risk than NBW control participants. For the ELBW group, each additional risk factor resulted in a 2‐point increase in internalizing scores, and two and a half times the odds of clinically significant internalizing symptoms (OR = 2.47, 95% CI = 1.63, 3.76). The protective effect of resiliency factors was also blunted among ELBW survivors.

Conclusions

Extremely low‐birth‐weight survivors may be more sensitive to traditional risk factors for psychopathology and less protected by resiliency factors. Intervention strategies aimed at preventing or reducing exposure to traditional childhood risk factors for psychopathology may reduce the burden of mental illness in adult survivors of prematurity.     

Outcome of patients with undifferentiated embryonal sarcoma of the liver treated according to European soft tissue sarcoma protocols

Background

To assess the outcomes of pediatric patients with undifferentiated embryonal sarcoma of the liver (UESL) and treatment including at least surgery and systemic chemotherapy.

Methods

This study included patients aged up to 21 years with a pathological diagnosis of UESL prospectively enrolled from 1995 to 2016 in three European trials focusing on the effects of surgical margins, preoperative chemotherapy, use of radiotherapy (RT), and chemotherapy.

Results

Out of 65 patients with a median age at diagnosis of 8.7 years (0.6–20.8), 15 had T2 tumors, and one had lymph node spread, 14 were Intergroup Rhabdomyosarcoma Study (IRS) I, nine IRS II, 38 IRS III, and four IRS IV. Twenty-eight upfront surgeries resulted in five operative spillages and 11 infiltrated surgical margins, whereas 37 delayed surgeries resulted in no spillages (p = .0119) and three infiltrated margins (p = .0238). All patients received chemotherapy, including anthracyclines in 47. RT was administered in 15 patients. With a median follow-up of 78.6 months, 5-year overall and event-free survivals (EFS) were 90.1% (95% confidence interval [CI]: 79.2–95.5) and 89.1% (95% CI: 78.4–94.6), respectively. Two out four local relapses had previous infiltrated margins and two out of three patients with metastatic relapses received reduced doses of alkylating agents. Infiltrated margins (p = .1607), T2 stage (p = .3870), use of RT (p = .8731), and anthracycline-based chemotherapy (p = .1181) were not correlated with EFS.

Conclusions

Multimodal therapy improved the outcome of UESL. Neoadjuvant chemotherapy for pediatric patients increases the probability of complete surgical resection. The role of anthracyclines and RT for localized disease remains unclear.     

Determining the prevalence of vestibular screening failures in pediatric cancer patients whose therapies include radiation to the head/neck and platin‐based therapies: A pilot study

1 Background

Sensorineural hearing loss due to ototoxic cancer therapy is well established; effects on the vestibular system are unknown. We examined the feasibility of implementing vestibular screens for pediatric cancer survivors exposed to ototoxic agents. The prevalence of screening failures is reported.

2 Methods

Cancer survivors who were 6–17 years, at least 1‐month posttreatment, and received ototoxic therapy (radiation to the head/neck, cisplatin, carboplatin) were eligible. Screening measures included (1) Pediatric Vestibular Symptom Questionnaire, (2) Modified Clinical Test of Sensory Interaction on Balance, and (3) Dynamic Visual Acuity.

3 Results

Vestibular screening failures were observed in 30 participants (60%). Patients with a brain tumor diagnosis were at increased risk for failures compared to nonbrain tumor patients (74.2% vs. 36.8%, P = 0.009). Patients who underwent brain surgery were at increased risk for failures compared to patients without brain surgery (71% vs. 42%, P = 0.043). Patients with a longer duration between end of treatment and vestibular screening had a reduced risk of failures, with an almost 20% decrease for each year between the time points (odds ratio = 0.812; 95% confidence interval: 0.683–0.964, P = 0.018). Receiving carboplatin correlated with a decreased risk of failure (P = 0.016), due to a negative correlation with other clinical risk factors (diagnosis of a brain tumor, major brain surgery) that are associated with vestibular screening failure.

4 Conclusion

Vestibular screening failures are highly prevalent in childhood cancer survivors who received ototoxic therapy. Broad screening of this population and further characterization of these patients are warranted.     

High‐risk age window for mortality in children with cystic fibrosis after lung transplantation

LTx in children with CF remains controversial. The UNOS database was queried from 1987 to 2013 for CF patients <18 yr of age at time of transplant. PCHR model was used to quantify hazard of mortality. 489 recipients were included in the survival analysis. The hazard function of post‐transplant mortality was plotted over attained age to identify age window of highest risk, which was 16–20 yr. Unadjusted PCHR model revealed ages immediately after the high‐risk window were characterized by lower hazard of mortality (HR = 0.472; 95% CI = 0.302, 0.738; p = 0.001). After adjusting for potential confounders, the decline in mortality hazard immediately after the high‐risk window remained statistically significant (HR = 0.394; 95% CI: 0.211, 0.737; p = 0.004). Hazard of mortality in children with CF after LTx was highest between 16 and 20 yr of attained age and declined thereafter.     

Rates and predictors of visits to primary care physicians during and after treatment of childhood acute lymphoblastic leukemia: A population-based cohort study

Introduction

Patient re-engagement with primary care physicians (PCPs) after cancer treatment is essential to facilitate survivorship care and to meet non-oncology primary care needs. We identified rates and predictors of PCP visits both during and after treatment among a population-based cohort of children with acute lymphoblastic leukemia (ALL).

Methods

Children of age less than 18 years at ALL diagnosis in Ontario between 2002 and 2012 were linked to administrative data and matched to controls without cancer. PCPs at diagnosis were identified and PCP visit rates during treatment compared between patients and controls. Post-treatment PCP visit rates were also calculated. Predictors included demographic-, disease-, and PCP-related variables.

Results

A total of 743/793 (94%) patients and 3112/3947 (79%) controls had a PCP at diagnosis. Almost half of patients (361/743, 45%) did not visit their PCP during treatment. Visit rate during treatment was 0.64 per person per year (PPPY) versus 1.4 PPPY among controls (adjusted rate ratio [aRR] 0.47, 95th confidence interval [95CI]: 0.40–0.54; p < .0001). No disease- or PCP-related factors were associated with visit rates. Total 711 patients completed frontline therapy; 287 (40.4%) did not have a PCP visit after treatment. Nonetheless, survivors overall visited PCPs post treatment more often than controls (aRR 1.4, 95CI: 1.2–1.6; p < .0001). Survivors who saw their PCP during treatment had post-treatment visit rates twice that of other survivors (aRR 2.0, 95CI: 1.6–2.5; p < .0001).

Conclusions

Only a portion of children with ALL see their PCPs during treatment and return to PCP care following treatment completion. Post-treatment engagement with PCPs may be improved by PCP involvement during ALL treatment.     

Localized vaginal/uterine rhabdomyosarcoma—results of a pooled analysis from four international cooperative groups

1 Background

Vaginal/uterine rhabdomyosarcoma (VU RMS) is one of the most favorable RMS sites. To determine the optimal therapy, the experience of four cooperative groups (Children's Oncology Group [COG], International Society of Pediatric Oncology (SIOP) Malignant Mesenchymal Tumor Group [MMT], Italian Cooperative Soft Tissue Sarcoma Group [ICG], and European pediatric Soft tissue sarcoma Study Group [EpSSG]) was analyzed.

2 Procedure

From 1981 to 2009, 237 patients were identified. Median age (years) at diagnosis differed by tumor location; it was 1.9 for vagina (n = 160), 2.7 for uterus corpus (n = 26), and 13.5 for uterus cervix (n = 51). Twenty‐eight percent of patients received radiation therapy (RT) as part of primary therapy (23% COG, 27% MMT, 46% ICG, and 42% EpSSG), with significant differences in the use of brachytherapy between the cooperative groups (23% COG, 76% MMT, 64% ICG, and 88% EpSSG).

3 Results

Ten‐year event‐free (EFS) and overall survival (OS) were 74% (95% CI, 67–79%) and 92% (95% CI, 88–96%), respectively. In univariate analysis, OS was inferior for patients with uterine RMS and for those with regional lymph node involvement. Although EFS was slightly lower in patients without initial RT (71% without RT vs. 81% with RT; P = 0.08), there was no difference in OS (94% without RT vs. 89% with RT; P = 0.18). Local control using brachytherapy was excellent (93%). Fifty‐one (51.5%) of the 99 survivors with known primary therapy and treatment for relapse were cured with chemotherapy with or without conservative surgery.

4 Conclusions

About half of all patients with VU RMS can be cured without systematic RT or radical surgery. When RT is indicated, modalities that limit sequelae should be considered, such as brachytherapy.     

Morbidity and healthcare use among mothers of children with cancer: A population-based study

Background

The impact of a child's cancer diagnosis on subsequent maternal physical health is unclear.

Methods

We identified all Ontario children diagnosed less than 18 years with cancer between 1992 and 2017. Linkage to administrative databases identified mothers who were matched to population controls. We identified physical health conditions, acute healthcare use, and preventive healthcare use through validated algorithms using healthcare data, and compared them between exposed (child with cancer) and unexposed mothers. Predictors of health outcomes were assessed among exposed mothers.

Results

We identified 5311 exposed mothers and 19,516 matched unexposed mothers. For exposed mothers, median age at last follow-up was 48 years, (interquartile range: 42–53). Exposed mothers had an increased risk of cancer (hazard ratio [HR] 1.2, 95% confidence interval [95% CI]: 1.0–1.5, p = .03), but not of any other adverse physical outcomes or of increased acute healthcare use. Exposed mothers were more likely to receive influenza vaccinations (odds ratio 1.4, 95% CI: 1.3–1.5, p < .0001), and underwent cancer screening at the same rate as unexposed mothers. Among exposed mothers, bereavement was associated with a subsequent increased risk of cancer (HR 1.7, 95% CI: 1.2–2.5, p = .004) and death (HR 2.2, 95% CI: 1.2–4.1, p = .01).

Conclusion

Mothers of children with cancer are at increased risk of developing cancer, but not of other adverse physical health outcomes, and were equally or more likely to be adherent to preventive healthcare practices. Bereaved mothers were at increased risk of subsequent cancer and death. Interventions targeting specific subpopulations of mothers of children with cancer or focused on screening for specific cancers may be warranted.     

The association of early linear growth and haemoglobin concentration with later cognitive,motor, and social–emotional development at preschool age in Ghana

It is important to identify the periods during childhood when exposure to environmental risk factors results in long‐term neurodevelopmental deficits. Stunting and anaemia may be sensitive indicators of exposure to such risks. In a prospective cohort enrolled before birth, we investigated the association of developmental scores at 4–6 years with (a) birth length and linear growth during three postnatal periods and (2) haemoglobin (Hb) concentration at three time points. Children were participants in a follow‐up study of a randomized controlled trial of nutritional supplementation in Ghana. At 4–6 years, cognitive, motor, and social–emotional developments were assessed using standard tests adapted for this population. We estimated the associations of length‐for‐age z‐score (LAZ) at birth and postnatal linear growth (n = 710) and Hb (n = 617) with developmental scores in regression models, using multistage least squares analysis to calculate uncorrelated residuals for postnatal growth. Cognitive development at 4–6 years was significantly associated with LAZ at birth (β = 0.12, 95% CI = 0.05, 0.19), ΔLAZ from 6 to 18 months (β = 0.16, 95% CI = 0.04, 0.28), and Hb at 18 months (β = 0.13, 95% CI = 0.06, 0.20), but not with ΔLAZ during 0–6 months, ΔLAZ from 18 months to 4–6 years, Hb at 6 months, or Hb at 4–6 years. No evidence of associations with motor or social–emotional development were found. These results suggest that in similar contexts, the earlier periods prior to birth and up to 18 months are more sensitive to risk factors for long‐term cognitive development associated with LAZ and Hb compared with later childhood. This may inform the optimal timing of interventions targeting improved cognitive development.     

Factors associated with syndemic anemia and stunting among children in Myanmar: A cross-sectional study from a positive deviance approach

BackgroundAnemia and stunting in children are detrimental to the prospects of a normal, healthy upbringing. Having similar risk factors and serious consequences, the syndemic aspect of these two ailments is mostly underrated, and positive deviant (PD) factors that ensure non-anemic status in stunted children have not been studied to date.MethodsThis study aimed to identify PD factors that have potential to prevent syndemic anemia among stunted children aged 6–59 months in Myanmar. This was a cross-sectional secondary analysis of the Myanmar Demographic and Health Survey (DHS) data conducted in 2016, applying the PD concept, where children who were stunted without anemia were considered as PDs.ResultsAmong 1248 stunted children, those who had the syndemic condition were compared with their PD peers in terms of maternal characteristics as well as socioeconomic and health-related factors. Multivariable logistic regression analyses were used to identify the determinants of syndemic state. The results showed that three out of every five stunted children were anemic. The syndemic risk was decreased among children of maternal age groups 20–34 years and 35–44 years: [aOR] = 0.19, 95% CI = 0.05–0.69; p = 0.012, and aOR = 0.19, 95% CI = 0.05–0.75; p = 0.018, respectively. Moderately stunted children (aOR = 0.53, 95% CI = 0.34–0.81; p = 0.004) and children who were not currently breastfed (aOR = 1.56, 95% CI = 1.01–2.41; p = 0.044) were less likely to develop the syndemic condition.ConclusionMaternal age, stunting severity, breastfeeding duration, and maternal anemic status are strong predictors in determining hemoglobin concentrations among stunted children. This study suggests that nutritional interventions targeting PD factors could represent syndemic action in improving child health.     

Healthcare utilization and cost barriers among U.S. childhood cancer survivors

Background

To evaluate healthcare utilization and cost barrier patterns among childhood cancer survivors (CCS) compared with noncancer controls.

Procedure

Using the 2014-2019 Behavioral Risk Factor Surveillance System, we identified CCS < 50 years and matched controls. We used chi-squared tests to compare characteristics between the two groups. Logistic regression analyses were used to assess the likelihood of having a checkup, receiving influenza vaccine, and experiencing healthcare cost barriers (being unable to see the doctor due to cost) during the past 12 months. Conditional models accounted for the matching.

Results

We included 231 CCS and 692 controls. CCS had lower household income (p < 0.001), lower educational attainment (p = 0.021), more chronic health conditions (p < 0.001), and a higher proportion of being current smokers (p = 0.005) than controls. Both groups had similar rates of having a checkup and influenza vaccine; however, a quarter of CCS experienced healthcare cost barriers compared with 13.9% in controls (p = 0.001; regression findings: adjusted odds ratio (aOR) = 1.72, 95% confidence interval (CI): 1.11-2.65). Compared with the youngest CCS group (18-24 years), CCS ages 25-29 years were five times more likely to experience healthcare cost barriers (aOR = 4.79; 95% CI, 1.39-16.54). Among CCS, current smokers were less likely to have a checkup (aOR = 0.46; 95% CI, 0.23-0.94). Uninsured CCS were less likely to have a checkup (aOR = 0.33; 95% CI, 0.14-0.75) and ∼8 times more likely to experience healthcare cost barriers (aOR = 8.28; 95% CI, 3.45-19.88).

Conclusion

CCS being 25-29 years, uninsured, or current smokers encounter inferior outcomes in healthcare utilization and cost barriers. We suggest emphasis on programs on care transition and smoking cessation for CCS.     

Long-term nephrotoxicity in irradiated pediatric kidney tumor survivors: A systematic review

Objective

Nephrotoxicity can occur as a side effect after treatment for kidney tumor in childhood. The use of radiotherapy (RT) has a potential additional effect.

Methods

A systematic electronic literature search that combined childhood kidney cancer with different treatments and nephrotoxicity terms was performed in EMBASE. Studies were included based on the reporting of nephrotoxicity occurrence after treatment for kidney tumor during pediatric age, with 75% of participants being under the age of 25 years at the time of diagnosis, and having been treated with any type of kidney surgery, chemotherapy, and/or RT.

Results

A pooled analysis did not show significant difference in estimated glomerular filtration rate between the group of patients who received RT compared with the group treated without RT (SMD −0.11 [95% CI −1.07–0.84] p = .733).

Conclusion

The current literature suggests that the use of RT does not have a significant impact on the decline of kidney function as independent factor.     

Second-line treatment of pediatric patients with relapsed rhabdomyosarcoma adapted to initial risk stratification: Data of the European Soft Tissue Sarcoma Registry (SoTiSaR)

Background

Outcome of relapsed disease of localized rhabdomyosarcoma remains poor. An individual treatment approach considering the initial systemic treatment and risk group was included in the Cooperative Weichteilsarkom Studiengruppe (CWS) Guidance.

Methods

Second-line chemotherapy (sCHT) ACCTTIVE based on anthracyclines (adriamycin, carboplatin, cyclophosphamide, topotecan, vincristine, etoposide) was recommended for patients with initial low- (LR), standard- (SR), and high-risk (HR) group after initial treatment without anthracyclines. TECC (topotecan, etoposide, carboplatin, cyclophosphamide) was recommended after initial anthracycline-based regimen in the very high-risk (VHR) group. Data of patients with relapse (n = 68) registered in the European Soft Tissue Sarcoma Registry SoTiSaR (2009–2018) were retrospectively analyzed.

Results

Patients of initial LR (n = 2), SR (n = 16), HR (n = 41), and VHR (n = 9) group relapsed. sCHT consisted of ACCTTIVE (n = 36), TECC (n = 12), or other (n = 15). Resection was performed in 40/68 (59%) patients and/or radiotherapy in 47/68 (69%). Initial risk stratification, pattern/time to relapse, and achievement of second complete remission were significant prognostic factors. Microscopically incomplete resection with additional radiotherapy was not inferior to microscopically complete resection (p = .17). The 5-year event-free survival (EFS) and overall survival (OS) were 26% (±12%) and 31% (±14%). The 5-year OS of patients with relapse of SR, HR, and VHR groups was 80% (±21%), 20% (±16%), and 13% (±23%, p = .008), respectively.

Conclusion

Adapted systemic treatment of relapsed disease considering the initial risk group and initial treatment is reasonable. New treatment options are needed for patients of initial HR and VHR groups.