Evaluating the use of parental reports to estimate health care resource utilization in children with suspected genetic disorders

Dragojlovic, Nick; Kim, Ellen; Elliott, Alison M.; Friedman, Jan M.; Lynd, Larry D.

doi:10.1111/jep.12876

Cited by 8 publications

(8 citation statements)

References 29 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The discrepancy between reports and records could have been due to either incomplete medical record ascertainment or over-reporting by mothers; we did not have records for visits at sites not affiliated with the primary HCP. Accuracy in parental recall of the type and frequency of health care service visits has been shown to be quite variable, with recall of hospitalizations generally considerably better than that of emergency department or outpatient visits [35]. In our sample, recall of a visit after one week was accurate in only 50% of four hospitalizations recorded by automated survey in the first year of life; this proportion was not statistically different from that for visits to emergency, urgent or primary care.…”

Section: Discussionmentioning

confidence: 61%

Recall accuracy of weekly automated surveys of health care utilization and infectious disease symptoms among infants over the first year of life

et al. 2019

View full text Add to dashboard Cite

Automated surveys, by interactive voice response (IVR) or email, are increasingly used for clinical research. Although convenient and inexpensive, they have uncertain validity. We sought to assess the accuracy of longitudinally-collected automated survey responses compared to medical records. Using data collected from a well-characterized, prospective birth cohort over the first year of life, we examined concordance between guardians’ reports of their infants’ health care visits ascertained by weekly automated survey (IVR or email) and those identified by medical chart review. Among 180 survey-visit pairs, concordance was 51%, with no change as number of visits per baby increased. Accuracy of recall was higher by email compared to IVR (61 vs. 43%; adjusted OR = 2.5 95% CI: 1.3–4.8), did not vary by health care encounter type (hospitalization: 50%, ER: 64%, urgent care: 44%, primary care: 52%; p = 0.75), but was higher for fever (77%, adjusted OR = 5.1 95%CI: 1.5–17.7) and respiratory illness (58%, adjusted OR = 2.9 95%CI: 1.5–5.8) than for other diagnoses. For the 75 mothers in these encounters, 69% recalled at least one visit; among 41 mothers with two or more visits, 85% recalled at least one visit. Predictors of accurate reporting by mothers after adjusting for illness in the baby included increased age and increased years of education (age per year, β = 0.05, p = 0.03; education per year, β = 0.08, p = 0.04). Additional strategies beyond use of automated surveys are needed to ascertain accurate health care utilization in longitudinal cohort studies, particularly in healthy populations with little motivation for accurate reporting.

show abstract

Section: Discussionmentioning

confidence: 61%

Recall accuracy of weekly automated surveys of health care utilization and infectious disease symptoms among infants over the first year of life

et al. 2019

View full text Add to dashboard Cite

show abstract

“…In prior work, we have shown that the sample of caregivers who completed the survey was broadly representative of the overall CAUSES sample, and that the risk of non-response bias is low. 29 Travel and time costs and estimated actual or potential cost savings from using TH are shown in Table 2. Average per-family total travel and time costs to visit C&W were Can$383 (SD = Can$631) for families who conducted in-person counselling through CAUSES as compared to Can$1043 (SD = Can$1177) for families who chose TH ( p = 0.008), while average costs to visit their local GP were Can$57 (SD = Can$106) and Can$78 (SD =Can $137), respectively ( p = 0.39).…”

Section: Resultsmentioning

confidence: 99%

“…chromosomal microarray analysis). [29][30][31][32][33] In addition to British Columbia, families residing in the province of Alberta and the Yukon Territory who were followed at BC Children's Hospital were invited to participate in the CAUSES Study. All families received pre-and post-test genetic counselling.…”

Section: Introductionmentioning

confidence: 99%

Utilization of telehealth in paediatric genome-wide sequencing: Health services implementation issues in the CAUSES Study

et al. 2021

Self Cite

View full text Add to dashboard Cite

Introduction Genome-wide sequencing (exome or whole genome) is transforming the care and management of paediatric patients with a rare disease because of its diagnostic capabilities. Genome-wide sequencing is most effective when both parents and the child are sequenced as a trio. Genetic counselling is recommended for all families considering genome-wide sequencing. Although telehealth is well established in genetic counselling for hereditary cancer and prenatal genetics, its use with genome-wide sequencing has not been well studied. The CAUSES Clinic at BC Children’s and Women’s Hospitals was a translational paediatric trio-based genome-wide sequencing initiative. Pre-test genetic counselling via telehealth (at a clinical site near the family’s residence) was offered to families who had been previously evaluated by a clinical geneticist. We report on the first 300 families seen in the CAUSES clinic and compare health services implementation issues of families seen via telehealth versus on-site. Methods Demographics, cost to families (travel and time), time to first appointment, complete trio sample accrual and diagnostic rates were studied. Results Of the 300 patients, 58 (19%) were seen via telehealth and 242 (81%) were seen on-site for pre-test counselling. The mean time to completion of accrual of trio samples in the telehealth group was 56.3 (standard deviation ±87.3) days versus 18.9 (standard deviation ±62.4) days in the onsite group ( p < 2.2 × 10−16). The mean per-family estimated actual or potential travel/time cost savings were greater in the telehealth group (Can$987; standard deviation = Can$1151) than for those seen on-site (Can$305; standard deviation = Can$589) ( p = 0.0004). Conclusions Telehealth allowed for access to genome-wide sequencing for families in remote communities and for them to avoid significant travel and time costs; however, there was a significant delay to accrual of the complete trio samples in the telehealth group, impacting on time of result reporting and delaying diagnoses for families for whom genome-wide sequencing was diagnostic.

show abstract

“…Secondly, we used data linkage to provide robust and comprehensive data on a large proportion of the healthcare costs, such as the hospital admissions and the utilization of healthcare resources listed under Medicare. When available, administrative data provide a reliable source of data compared to study participants’ recollection of events [ 3 ]. Data such as that collected in our study are important for capturing the wide-ranging healthcare and societal costs associated with familial ID in order to understand the broad financial impact on families and the government.…”

Section: Discussionmentioning

confidence: 99%

“…The severity of familial ID is classified according to the intelligence quotient (IQ) of the affected person, as follows: mild (IQ 50-69), moderate (35)(36)(37)(38)(39)(40)(41)(42)(43)(44)(45)(46)(47)(48)(49), severe (20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30)(31)(32)(33)(34), and profound (<20) [2]. The global prevalence of ID is approximately 1%, with an estimated 20-50% of all ID cases of being of genetic etiology, and 450 identified genes are known to cause ID [3]. In Australia, roughly 450,000 (1.8%) of the population was estimated to have ID in 2021 [4].…”

Section: Introductionmentioning

confidence: 99%

The Healthcare and Societal Costs of Familial Intellectual Disability

Schofield,

Shrestha,

Tan

et al. 2024

IJERPH

View full text Add to dashboard Cite

Most of the studies on the cost of intellectual disability are limited to a healthcare perspective or cohorts composed of individuals where the etiology of the condition is a mixture of genetic and non-genetic factors. When used in policy development, these can impact the decisions made on the optimal allocation of resources. In our study, we have developed a static microsimulation model to estimate the healthcare, societal, and lifetime cost of individuals with familial intellectual disability, an inheritable form of the condition, to families and government. The results from our modeling show that the societal costs outweighed the health costs (approximately 89.2% and 10.8%, respectively). The lifetime cost of familial intellectual disability is approximately AUD 7 million per person and AUD 10.8 million per household. The lifetime costs to families are second to those of the Australian Commonwealth government (AUD 4.2 million and AUD 9.3 million per household, respectively). These findings suggest that familial intellectual disability is a very expensive condition, representing a significant cost to families and government. Understanding the drivers of familial intellectual disability, especially societal, can assist us in the development of policies aimed at improving health outcomes and greater access to social care for affected individuals and their families.

show abstract

Evaluating the use of parental reports to estimate health care resource utilization in children with suspected genetic disorders

Abstract: Parental surveys are a valuable tool for estimating health care resource utilization during a 6-month recall period for children with suspected genetic disorders but are best used to complement utilization data collected from other sources.

Cited by 8 publications

References 29 publications

Recall accuracy of weekly automated surveys of health care utilization and infectious disease symptoms among infants over the first year of life

Recall accuracy of weekly automated surveys of health care utilization and infectious disease symptoms among infants over the first year of life

Utilization of telehealth in paediatric genome-wide sequencing: Health services implementation issues in the CAUSES Study

The Healthcare and Societal Costs of Familial Intellectual Disability

Contact Info

Product

Resources

About