Acute lymphoblastic leukemia (ALL) is the most common childhood malignancy and among the most common malignancies in young adults and requires a unique pattern of healthcare utilization including an ...acute/emergent presentation and an intensive initial 8 months of therapy followed by two years of outpatient treatment. The COVID-19 pandemic caused massive global disruptions in healthcare use and delivery. This report aims to examine the effects of the COVID-19 pandemic on the presentation, diagnosis and continued management of childhood and young adult ALL in regard to utilization and cost of care among commercially insured individuals in the United States.
Utilizing a commercial insurance claims database, 529 pediatric and young adult patients were identified who were diagnosed with ALL between January 2016 and March 2021. New diagnoses were evaluated by era and demographics. Utilization was measured by COVID-related era as number of inpatient and outpatient encounters, inpatient days, and cumulative cost during the initial 8 months of therapy. None of these cost or utilization factors changed significantly during or shortly after the pandemic. These findings reinforce that the necessary care for pediatric and young adult ALL was unwavering despite the massive shifts in the healthcare system caused by the COVID-19 pandemic. This provides a valuable benchmark as we further examine the factors that influence the pandemic's impact on health equity and access to care, especially in vulnerable pediatric and young adult populations. This is the first investigation of the effect of the COVID-19 pandemic on utilization and cost of care in pediatric and young adult cancer.
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IZUM, KILJ, NUK, PILJ, PNG, SAZU, UL, UM, UPUK
Background
B‐lineage acute lymphoblastic leukemia (B‐ALL) is the most common malignancy of childhood. With the introduction of novel cellular therapies, cost of care is a critical component and the ...financial burden experienced by patients and society requires evaluation.
Aims
This study aims to assess the utilization and cost of care for chimeric antigen receptor T‐cell (CAR‐T) therapy for pediatric ALL patients with commercial insurance coverage in the United States.
Methods and Results
Using de‐identified commercial insurance data from the OptumLabs® Data Warehouse, a cohort of 37 patients, aged 1‐25 years, with B‐ALL treated with CAR‐T therapy between Oct 2016 and Dec 2021 in the United States was identified. Cost was evaluated for a 90 day period encompassing CAR‐T infusion and by administration and complication characteristics. Among the 37 identified B‐ALL patients that received a CAR‐T product infusion, 14 patients were female, median age at administration was 13 years. The median 90‐day total cost was $620,500 (Mean: $589,108). Inpatient cost accounted for approximately 71% of the total cost with an average of 28 inpatient days per patient. Although inpatient cost was slightly higher in the older age group (aged 10‐25 years) and in patients with a code for cytokine release syndrome (CRS), these differences were not statistically significant.
Conclusion
This real‐world cost analysis shows for the first time the encompassing cost of CAR‐T therapy for pediatric B‐ALL patients in the US with commercial insurance. This study provides a valuable benchmark that can be used to analyze the financial implications of CAR‐T therapy for pediatric B‐ALL therapy on health systems.
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FZAB, GIS, IJS, KILJ, NLZOH, NUK, OILJ, SBCE, SBMB, UL, UM, UPUK
OBJECTIVELong-term psychological impacts are well--documented among childhood cancer survivors. To our knowledge, however, no research has been conducted to investigate obsessive--compulsive and ...related disorders (OCRD) among childhood -cancer survivors (CCS).METHODSUsing a large electronic medical record database, relative risk were calculated to examine associations between demographic characteristics and childhood cancer type and OCRDs among childhood cancer survivors.RESULTSAmong 121 survivors of childhood cancer diagnosed with OCRD, 57% were female. The most common childhood cancer diagnoses were leukemia/lymphoma (41%) and central nervous system (CNS) malignancies (38%), and OCRD diagnoses most frequently observed were obsessive-compulsive disorder (OCD; 76%) and excoriation disorder (13%). Female sex (RR= 1.39, 95% confidence interval (CI) 1.17-1.61), White race (RR= 1.28, 95% CI 1.15-1.36) and history of CNS malignancies (RR= 1.36, 95% CI 1.18, 1.92) were associated with OCD.CONCLUSIONSNumerous factors, including sex, race, and cancer type, were seen as contributors to risk variance for OCRDs, particularly OCD, among CCS, compared to CCS with no OCRD diagnosis. This provides an enhanced understanding of risk factors for OCRD development and may help improve early identification and care for at-risk survivors.
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BFBNIB, NUK, PILJ, SAZU, UL, UM, UPUK
To investigate the temporal trends and factors associated with outpatient rehabilitation utilization and costs for pediatric acute lymphoblastic leukemia (ALL).
Deidentified administrative claims ...data and longitudinal health information on patients representing a mixture of ages, ethnicities, and geographic regions across the United States were accessed using Optum Labs Data Warehouse. Regression models were constructed to assess associations of outpatient rehabilitation with age, sex, race and ethnicity, year of diagnosis, and region.
Outpatient rehabilitation.
1000 Patients aged 1-30 years with a new diagnosis of ALL between 1993 and 2017 and continuous insurance coverage (N=1000).
Not applicable.
Outpatient rehabilitation service utilization and cost based on reimbursed charge codes, summarized over 36 months after cancer diagnosis.
In 1000 patients, utilization of outpatient rehabilitation services increased from 20% in 1993-2002 to 55% in 2013-2017. In the earliest era examined, physical and/or occupational therapy was provided to 18% and increased to 54% in the latest years. Speech service utilization remained between 5%-8% across timepoints. Inflation-adjusted cost for provision of services did not change significantly across time and remained low, accounting for a median of 1.3% (Q1, Q3 0.3, 3.4) of total treatment cost in 1993-2002 and decreasing to a median 0.4% (Q1, Q3, 0.1, 1.0) in 2013-2017. Age 1 to 5 years at ALL diagnosis was associated with increased rehabilitation visit number and cost, and treatment in the Midwest was associated with increased likelihood of outpatient rehabilitation service utilization compared to other geographic regions.
Outpatient rehabilitation services are being increasingly provided to patients with ALL at a relatively low cost per patient, yet geographic variability in care utilization is evident. These services do not add excessively to the overall cost of leukemia care and thus cost containment should not be an excuse to limit access.
Quantifying secular variations in the chemical composition of the Martian crust provides unique insights into the processes that have guided the evolution of the Martian crust‐mantle system. Using ...global abundances for a suite of elements determined by the Gamma Ray Spectrometer (GRS) on board the Mars Odyssey spacecraft and global mapping of apparent surface age adapted from existing geologic maps in the USGS Martian Geologic Investigation series, we report the average abundance of K, Th, Fe, Cl, H, and Si for the major Martian geologic epochs (Noachian, Hesperian, and Amazonian). Average GRS‐determined K and Th abundances generally decrease by 9% and 7%, respectively, between the Hesperian and the Amazonian, possibly implying evolving magma chemistry throughout major resurfacing events (although the effects of surficial alteration processes cannot be entirely discounted). GRS‐determined Fe and Cl averages increase by 12% and 19%, respectively, with younger apparent relative surface age, suggesting the possible mobilization and transport of these elements through aqueous processes (although an igneous origin for the variation in Fe also cannot be excluded). While H abundance does vary with surface age, the relationship is likely not governed by geologic processes. No statistically reliable apparent surface age relation was found for Si.
Introduction: Acute lymphoblastic leukemia (ALL) is the most common malignancy of childhood, with a current incidence of 3,000 cases annually. Incremental alterations in treatment over time have ...resulted in overall survival rates that now exceed 90%. Although minor differences exist between ALL treatment protocols from different cooperative groups, risk-stratified treatment for ALL is generally standard across the country. As more has been understood about ALL biology and outcomes, treatments have been further risk stratified to optimize outcomes for higher risk individuals while minimizing toxicities for those with more favorable disease characteristics. With the improvements in outcome for ALL, cost of care is a critical component and financial toxicity for patients and society requires evaluation.
Methods: Using commercial insurance data from the OptumLabs ® Data Warehouse, a cohort of 1941 ALL patients was identified, aged 1-30 years and diagnosed between 2000 and 2017 in the United States. ALL diagnosis was confirmed based on ICD-9 and ICD-10 diagnostic codes, in combination with bone marrow and lumbar puncture procedure codes within 14 days of diagnosis. Individuals were followed up to 36 months from diagnosis. Mean monthly and cumulative costs were computed for the initial 8 months of therapy and for 36 months from diagnosis, as were mean number of inpatient and outpatient days. Costs were further broken down by the types of services provided. All results were stratified by age group (1-9 years, 10-12 years, ≥13 years) as a proxy for risk group, with the younger group most consistent with standard risk disease and the older groups indicating high risk and very high risk disease, respectively.
Results: Among the 1941 identified ALL patients (1-9 years, 1233; 10-12 years, 162; ≥13 years, 546), 44% were female, 51% were white, median age at diagnosis was 6 years (interquartile range 3-14 years) and 941 of enrolled patients had continuous data through 36 months. Mean cost of care over the approximate 3 years of therapy was $537,000, with approximately 58% of costs incurred within the initial intensive 8 months of therapy. Mean cost was highest among the ≥13 year age group, both for the initial 8 months of therapy ($487,000, 95% CI $449,300-530,400)), as well as over the 36 months following diagnosis ($850,000, 95% CI $778,400-937,100) (Figure). This was reflective of the highest mean number of inpatient days among the ≥13 year age group (88.1 days over 36 months, 95% CI 82.1-94.9) compared to the 10-12 year group (73.2 days, 95% CI 64.3-85.1) and the 1-9 year group (47.5 days, 95% CI 45.2-50.9). Mean number of outpatient visits over 36 months was not significantly different between 1-9 and 10-12 year age groups but was significantly higher among the ≥13 year age group compared with the 1-9 year group. Thirty-six month survival varied by age group and was highest among the 1-9 year group (95.9%, 95% CI 94.6-97.2) compared with the 10-12 year group (90.5%, 95% CI 85-4-95.8) and the ≥13 year group (79.6%, 95% CI 75.8-83.6).
Conclusions: This robust real-world cost analysis shows for the first time that the average cost of ALL care in the current treatment era, including inpatient and outpatient data, is more than $500,000. We showed that the cost of care is highest among the oldest pediatric age group, indicating increasing cost with increased therapeutic intensity. This study provides a valuable benchmark that can be used to analyze the financial toxicity of ALL therapy on patients and families and can be re-assessed over time as novel therapeutics are introduced into ALL therapy.
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No relevant conflicts of interest to declare.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NLZOH, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UILJ, UL, UM, UPCLJ, UPUK, ZAGLJ, ZRSKP
OBJECTIVEThe aims of this study were to describe the use of Ringerʼs lactate (LR) or normal saline (NS) for resuscitation among children with diabetic ketoacidosis (DKA) and compare the effect of ...fluid type on cost, length of stay, and rate of cerebral edema (CE).
METHODSThis is a retrospective study of 49,737 children aged 0 to 17 years with DKA between January 1, 2005, and September 30, 2015, using data from the Pediatric Health Information System. Treatment with LR or NS was identified. Our primary outcomes were total adjusted cost and length of stay. Our secondary outcome was CE rate per 1000 episodes.
RESULTSThe majority of patients were treated with NS (n = 43,841 88%) compared with LR (n = 1762 4%) or both NS and LR (n = 4134 8%). Hospital-year–specific practice patterns were used to investigate the effect of fluid type across resuscitation fluid groups. Total adjusted cost was $1160 less (95% confidence interval, −1468 to −852), or −14.2%, for cases with any episode of LR compared with NS only. Length of stay was not different across groups. The rate of cerebral edema per 1000 episodes was 12.7 for cases with any episode of LR compared with 34.6 NS only (difference, −21.9; 95% confidence interval, −30.4 to −13.3).
CONCLUSIONSRingerʼs lactate was infrequently used for resuscitation of pediatric DKA patients. However, resuscitation with LR compared with NS was associated with lower total cost and rates of CE. Further investigation using patient-level clinical and laboratory data is needed to evaluate factors that drive cost and risk of CE development with each fluid.
Background
Pleuropulmonary blastoma (PPB) is the most common lung cancer of infancy and early childhood. Type I PPB is a purely cystic lesion that has a microscopic population of primitive small ...cells with or without rhabdomyoblastic features and may progress to type II or III PPB, whereas type Ir lacks primitive small cells.
Methods
Children with suspected PPB were enrolled in the International PPB/DICER1 Registry. Pathology was centrally reviewed, and follow‐up was ascertained annually.
Results
Between 2006 and 2022, 205 children had centrally reviewed type I or Ir PPB; 39% of children with type I and 5% of children with type Ir PPB received chemotherapy. Outcomes were favorable, although 11 children (nine with type I and two with type Ir PPB) experienced progression to type II/III (n = 8) or regrowth of type I PPB at the surgical site (n = 3), none of whom received chemotherapy before progression. Age and cyst size in combination were more suitable than either factor alone in predicting whether a particular lesion was type I or Ir PPB.
Conclusions
For young children with type I PPB, outcomes are favorable, but complete resection is indicated because of the risk for progression. Chemotherapy may be useful in a subset of children at increased risk for recurrence/progression. Efforts to risk stratify children with type I PPB to optimize outcomes while reducing treatment‐related side effects are underway.
Outcomes for children with type I and Ir pleuropulmonary blastoma (PPB) are favorable, although progression to advanced disease is noted in some children. Chemotherapy may be useful in a subset of children with type I PPB who are at increased risk for recurrence/progression.
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BFBNIB, FZAB, GIS, IJS, KILJ, NLZOH, NUK, OILJ, SBCE, SBMB, UL, UM, UPUK
Since the original description of pathogenic germline DICER1 variation underlying pleuropulmonary blastoma (PPB), the spectrum of extrapulmonary neoplasms known to be associated with DICER1 has ...continued to expand and now includes tumors of the ovary, thyroid, kidney, eye, and brain among other sites. This report documents our experience with another manifestation: a primitive sarcoma that resembles PPB and DICER1-associated sarcoma of the kidney. These tumors are distinguished by their unusual location in the peritoneal cavity, associated with visceral and/or parietal mesothelium. A total of seven cases were identified through pathology review in children presenting at a median age of 13 years (range 3-14 years). Primary sites of origin included the fallopian tube (four cases), serosal surface of the colon (one case), and pelvic sidewall (two cases). One case had pathologic features of type I PPB, another type Ir (regressed) PPB, and the remaining five had features of type II or III PPB with a mixed primitive sarcomatous pattern with or without cystic elements. All had a pathogenic DICER1 variation identified in germline and/or tumor DNA. PPB-like peritoneal tumors represent a newly described manifestation of DICER1 pathogenic variation whose pathologic features are also recapitulated in DICER1-related renal sarcoma, cervical embryonal rhabdomyosarcoma, and some Sertoli-Leydig cell tumors with heterologous elements. Tumors arising from the fallopian tube or elsewhere in the abdomen/pelvis, especially those with heterogeneous rhabdomyosarcomatous and/or cartilaginous differentiation, should prompt consideration of germline and tumor DICER1 testing.
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GEOZS, IJS, IMTLJ, KILJ, KISLJ, NLZOH, NUK, OILJ, PNG, SAZU, SBCE, SBJE, UILJ, UL, UM, UPCLJ, UPUK, ZAGLJ, ZRSKP
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