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

被引：5

作者：

Baker, Emma K. ^{[1
,2
,3
]}

Arora, Sheena ^{[4
]}

Amor, David J. ^{[2
,5
]}

Date, Perrin ^{[1
,11
]}

Cross, Meagan ^{[6
]}

O'Brien, James ^{[7
]}

Simons, Chloe ^{[6
]}

Rogers, Carolyn ^{[8
]}

Goodall, Stephen ^{[4
]}

Slee, Jennie ^{[9
]}

Cahir, Chris ^{[10
]}

Godler, David E. ^{[1
,2
]}

机构：

[1] Royal Childrens Hosp, Murdoch Childrens Res Inst, Diag & Dev, 50 Flemington Rd, Parkville, Vic 3052, Australia

[2] Univ Melbourne, Dept Paediat, Fac Med Dent & Hlth Sci, Parkville, Vic, Australia

[3] La Trobe Univ, Sch Psychol & Publ Hlth, Bundoora, Vic, Australia

[4] Univ Technol Sydney, Ctr Hlth Econ Res & Evaluat, Broadway, NSW, Australia

[5] Royal Childrens Hosp, Murdoch Childrens Res Inst, Neurodisabil & Rehabil, Parkville, Vic, Australia

[6] Fdn Angelman Syndrome Therapeut FAST, Salisbury, Qld, Australia

[7] Prader Willi Syndrome Australia Ltd, Melbourne, Vic, Australia

[8] Hunter Genet, Genet Learning Disabil Serv, Waratah, NSW, Australia

[9] Govt Western Australia, Genet Serv Western Australia, Dept Hlth, Perth, WA, Australia

[10] Dup15q Australia Ltd, Melbourne, Vic, Australia

[11] La Trobe Univ, Olga Tennison Autism Res Ctr, Melbourne, Vic, Australia

来源：

JOURNAL OF AUTISM AND DEVELOPMENTAL DISORDERS | 2023年 / 53卷 / 04期

基金：

英国医学研究理事会; 澳大利亚国家健康与医学研究理事会;

关键词：

Fragile X syndrome; Angelman syndrome; Prader-Willi syndrome; Chromosome 15 Duplication syndrome; Health Economics; PRADER-WILLI-SYNDROME; ABERRANT BEHAVIOR CHECKLIST; AUTISM SPECTRUM DISORDER; ANGELMAN-SYNDROME; CHILDREN; FAMILIES; BURDEN;

D O I：

10.1007/s10803-021-05193-4

中图分类号：

B844 [发展心理学（人类心理学）];

学科分类号：

040202 ;

摘要：

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.

引用

页码：1682 / 1692

页数：11

共 51 条

[1]

AMAN MG, 1985, AM J MENT DEF, V89, P485

[2]

[Anonymous], 2019, CONS PRIC IND

[3] Societal cost of childhood intellectual disability in Australia [J].

Arora, S. ;

Goodall, S. ;

Viney, R. ;

Einfeld, S. .

JOURNAL OF INTELLECTUAL DISABILITY RESEARCH, 2020, 64 (07) :524-537

[4] Intragenic DNA methylation in buccal epithelial cells and intellectual functioning in a paediatric cohort of males with fragile X [J].

Arpone, Marta ;

Baker, Emma K. ;

Bretherton, Lesley ;

Bui, Minh ;

Li, Xin ;

Whitaker, Simon ;

Dissanayake, Cheryl ;

Cohen, Jonathan ;

Hickerton, Chriselle ;

Rogers, Carolyn ;

Field, Mike ;

Elliott, Justine ;

Aliaga, Solange M. ;

Ling, Ling ;

Francis, David ;

Hearps, Stephen J. C. ;

Hunter, Matthew F. ;

Amor, David J. ;

Godler, David E. .

SCIENTIFIC REPORTS, 2018, 8

[5]

Australian Bureau of Statistics, 2019, AV WEEKL EARN AUSTR

[6]

Australian Bureau of Statistics, 2018, EMPL EARN HOURS AUST

[7]

Australian Government, 2019, NAT DIS INS SCHEM SU

[8]

Australian Government Department of Education Skills and Employment, 2020, WHAT IS AUSTR GOV DO

[9]

Australian Government Department of Health, 2020, PHARM BEN SCHEM

[10]

Australian Government Department of Health, 2019, MEDICARE BENEFITS SC

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