Validity of a condition specific outcome measure for fragile X syndrome: the Aberrant Behaviour Checklist‐utility index

Raspa, Melissa; Sacco, Patricia; Candrilli, Sean; Bishop, Ellen; Petrillo, Jennifer

doi:10.1111/jir.12264

Cited by 6 publications

(4 citation statements)

References 49 publications

(66 reference statements)

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“…In fact, once behavioral and emotional problems were covaried, our results showed that families with FXS experienced a greater negative impact on feelings about parenting, finances, siblings, and the degree of difficulty of living with a child or adolescent with FXS. In general, our results are similar to those of other studies (Haessler et al, 2016;Johnston et al, 2003;Lewis et al, 2006;Raspa et al 2016).…”

Section: Article In Presssupporting

confidence: 93%

“…In the study by Johnston et al (2003), 25% of mothers of children with FXS felt stress due to isolation and lack of parental competence. Problems and concerns about parenting and family relationships are affected by the diagnostic status of the child, and are greater in the case of parents of children with FXS (Lewis et al, 2006;Raspa et al, 2016).…”

Section: Article In Pressmentioning

confidence: 99%

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Impact of Fragile X Syndrome on Their Families

Iriarte-Redín

Ferrer

Hidalgo-Alés

2020

Psicología Educativa

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The aim of the present study is to describe the behavioral and emotional disturbances in the Fragile X Syndrome (FXS) and to analize the impact of having a member with FXS on different aspects of family functioning. A sample of 79 parents with a member with FXS and 80 parents with a member with typical development participated in the study. Results showed that between 17% and 66% of children and adolescents with FXS displayed significant behavioral and emotional disturbances and fewer prosocial behaviors. In addition, after controlling for behavioral and emotional difficulties, our results showed that families affected by FXS experienced a more negative impact on feelings about parenting, finances, siblings, and the degree of difficulty of living with a child or adolescent with FXS, and greater family adaptability and cohesion than the comparison group. In conclusion, our results highlight that it is important to consider these differences in clinical practice when supporting and counselling families affected by FXS.

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Section: Article In Presssupporting

confidence: 93%

Section: Article In Pressmentioning

confidence: 99%

Impact of Fragile X Syndrome on Their Families

Iriarte-Redín

Ferrer

Hidalgo-Alés

2020

Psicología Educativa

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“…Further financial burden was caused by home and car adaptations and the costs of additional healthcare not covered by public healthcare provision, such as physiotherapy, speech therapy, and occupational therapy [24]. Many families were forced to have at least one caregiver (e.g., mother or father) change their job or working hours, and 35% to 45% of families reported that a caregiver had to leave their job entirely [30,[36][37][38]. Seizures in FXS have been shown to be associated with caregiver financial burden and quitting work [32].…”

Section: Impact On Work and Productivitymentioning

confidence: 99%

Impact of developmental and epileptic encephalopathies on caregivers: A literature review

Gallop

Lloyd

Olt

et al. 2021

Epilepsy & Behavior

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Objectives: Developmental and epileptic encephalopathies (DEEs) are rare neurodevelopmental disorders characterized by early-onset seizures and numerous comorbidities. Due to the complex requirements for the care of a child with a DEE, these disorders would be expected to impact health-related quality of life (HRQL) for caregivers as well as for patients. The objective of this literature review was to describe the impact of DEEs on the HRQL, emotional wellbeing, and usual activities (social, work, relationships, etc.) of caregivers, including the wider impact on other family members such as siblings. Methods: A literature search was conducted in May 2020 using MEDLINE Ò and Embase Ò databases. Quantitative and qualitative studies were identified using search terms related to family, disease type (including >20 specific DEEs), and quality of life/methodology. Each study was assessed for relevance and was graded using customized critical appraisal criteria. Findings from studies that were given the highest quality ratings were summarized and used to develop a conceptual model to illustrate the complex impact of DEEs on caregiver HRQL. Results: Sixty-seven relevant studies were identified, of which 39 (27 quantitative, 12 qualitative) met the highest appraisal criteria. The studies recruited caregivers of patients with one of eight individual DEEs, or pediatric intractable or refractory epilepsy. Most studies reported negative impacts on HRQL and emotional wellbeing in caregivers. The wide-ranging impact of a DEE was highlighted by reports of negative effects on caregivers' physical health, daily activities, relationships, social activities, leisure time, work, and productivity. Factors that influenced the perceived impact included demographic characteristics (e.g., child's age, living arrangements, family income) and clinical factors (e.g., feeding or sleep difficulties, disease severity). Few studies evaluated the impact on siblings. Conclusions: There is evidence that DEEs can impact HRQL and emotional wellbeing and can limit usual activities for the primary caregiver and their wider family. However, no research was identified regarding many individual DEEs, and only limited research assessed the impact on different family members with most studies focusing on mothers. Further research is required to understand the influence of certain factors such as the age of the patient, disease severity, and seizures on caregiver burden. Furthermore, the review highlighted the lack of appropriate measurement tools to assess caregiver HRQL in this population.

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“…Twenty-three unique valuation methods were identified. These were grouped into six key categories: (i) VAS—EQ-5D VAS (number of samples = 348; 8.8%), EQ-5D-Y VAS ( n = 232; 5.8%) and stand-alone VAS ( n = 252; 6.3%); (ii) trade-off-based direct valuation methods—TTO ( n = 171; 4.3%), SG ( n = 227; 5.7%), chained gamble and adjusted SG ( n = 143; 3.6%); (iii) adult-specific MAUIs—EQ-5D ( n = 424; 10.7%), SF-6D ( n = 34; 0.9%), AQoL-5D ( n = 16; 0.4%) and 15D ( n = 2; 0.05%); (iv) MAUIs compatible with both childhood and adult populations—QWB ( n = 224; 5.6%), HUI2 ( n = 482; 12.1%), HUI3 ( n = 822; 20.7%), modified HUI (10-dimension variant of HUI [ 41 ], HUI3 with ‘worst imaginable health’ as 0 instead of death [ 42 ]; n = 8; 0.2%) and ABC-UI (Aberrant Behaviour Checklist Utility Index) [ 43 ] ( n = 1; 0.03%); (v) childhood-specific MAUIs—EQ-5D-Y ( n = 108; 2.7%), CHU9D ( n = 231; 5.8%), 16D ( n = 73; 1.8%), 17D ( n = 39; 1.0%), AQoL-6D ( n = 50; 1.3%), PAHOM (Pediatric Asthma Health Outcome Measure) [ 44 ] ( n = 69; 1.7%) and CH-6D (Child Health-6 Dimensions) [ 45 ] ( n = 3; 0.08%);and (vi) mapping non-preference-based clinical measures to utility indices [ 46 – 50 ] ( n = 15; 0.4%).…”

Section: Resultsmentioning

confidence: 99%

Patterns, trends and methodological associations in the measurement and valuation of childhood health utilities

et al. 2019

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Purpose To systematically assess patterns and temporal changes in the measurement and valuation of childhood health utilities and associations between methodological factors. Methods Studies reporting childhood health utilities using direct or indirect valuation methods, published by June 2017, were identified through PubMed, Embase, Web of Science, PsycINFO, EconLit, CINAHL, Cochrane Library and PEDE. The following were explored: patterns in tariff application; linear trends in numbers of studies/samples and paediatric cost–utility analyses (CUAs) and associations between them; changes in proportions of studies/samples within characteristic-based categories over pre-specified periods; impact of National Institute for Health and Care Excellence (NICE) guidance on primary UK research and associations between valuation method, age and methodological factors. Results 335 studies with 3974 samples covering all ICD-10 chapters, 23 valuation methods, 12 respondent types and 42 countries were identified by systematic review. 34.0% of samples using indirect methods compatible with childhood applied childhood-derived tariffs. There was no association between numbers of studies/samples and numbers of CUAs. Compared to 1990–2008, 2009–June 2017 saw a significant fall in the proportion of studies using case series; significant compositional changes across ICD-10 chapters and significantly higher sample proportions using childhood-specific and adult-specific indirect valuation methods, and based on pre-adolescents, self-assessment, self-administration and experienced health states. NICE guidance was weakly effective in promoting reference methods. Associations between valuation method, age and methodological factors were significant. Conclusion 1990–2017 witnessed significant changes in primary research on childhood health utilities. Health technology assessment agencies should note the equivocal effect of methodological guidance on primary research. Electronic supplementary material The online version of this article (10.1007/s11136-019-02121-z) contains supplementary material, which is available to authorized users.

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Validity of a condition specific outcome measure for fragile X syndrome: the Aberrant Behaviour Checklist‐utility index

Cited by 6 publications

References 49 publications

Impact of Fragile X Syndrome on Their Families

Impact of Fragile X Syndrome on Their Families

Impact of developmental and epileptic encephalopathies on caregivers: A literature review

Patterns, trends and methodological associations in the measurement and valuation of childhood health utilities

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