The Cost of Raising Individuals with Fragile X or Chromosome 15 Imprinting Disorders in Australia

Abstract

The study characterised differences in costs associated with raising a child between four rare disorders and examined the associations between these costs with clinical severity. Caregivers of 108 individuals with Prader-Willi, Angelman (AS), Chromosome 15q Duplication and fragile X (FXS) syndromes completed a modified Client Services Receipt Inventory and participants completed intellectual/developmental functioning and autism assessments. AS incurred the highest yearly costs per individual ($AUD96,994), while FXS had the lowest costs ($AUD33,221). Intellectual functioning negatively predicted total costs, after controlling for diagnosis. The effect of intellectual functioning on total costs for those with AS was significantly different to the other syndromes. The study highlights the significant costs associated with these syndromes, particularly AS, linked with severity of intellectual functioning.

Estimating the impact of Angelman syndrome on parental productivity in Australia using productivity-adjusted life years

“…Furthermore, limitations in the granularity of data available to support the estimation of the parental productivity index meant that the parental productivity impact associated with the 'lengthy diagnostic odyssey' commonly experienced by the families of persons with AS was not to be estimated (Wheeler et al, 2017). However, it is anticipated that diagnostic approaches which support reducing the time to an AS diagnosis could have a flow-on benefit of reducing a component of the parental stress associated with having a child with AS (Baker et al, 2021;Doble et al, 2020). This is the first known study to estimate the total impact of caring for a child with AS on parental productivity, as well as the first study known to estimate the PALYs lost by a parental or caregiver population.…”

“…However, these studies each addressed slightly different research questions, and hence their results are not directly comparable to those described in the present study. Baker et al (2021) estimated the productivity losses arising from the under- and un-employment of individuals with AS from an individual and government perspective. In doing so, Baker et al (2021) found that the mean income tax revenue lost due to the reduced employment of individuals with AS totalled AUD$19,367 (95% CI: $7,785-$30,949) per annum.…”

“…Baker et al (2021) estimated the productivity losses arising from the under- and un-employment of individuals with AS from an individual and government perspective. In doing so, Baker et al (2021) found that the mean income tax revenue lost due to the reduced employment of individuals with AS totalled AUD$19,367 (95% CI: $7,785-$30,949) per annum. By contrast, Arora et al (2020) sought to quantify the costs associated with intellectual disability in childhood in Australia, including costs associated with healthcare, informal care, and productivity losses.…”

“…The model structure is depicted in Figure S1. Key data were obtained from a prospective cohort of families of children with AS who participated in a natural history study of chromosome 15 imprinting disorders (Baker et al, 2018(Baker et al, , 2021, peer-reviewed literature, and publicly-available data reported by the Australian Bureau of Statistics (ABS). The data analyses and supporting assumptions are discussed in the following sections, with further detail provided in Tables S1-2.…”

“…Many of these factors mean that children with AS, and their families, are often subject to a 'lengthy diagnostic odyssey' with most only receiving a diagnosis between the ages of one and five years (Wheeler et al, 2017). Arguments for more systematic diagnostic efforts for AS have been made elsewhere (Baker et al, 2021).…”