Evidence map›Paper›PMID 27622144›Full record

ArticleMolecular genetics and metabolism reports2016

The personal burden for caregivers of children with phenylketonuria: A cross-sectional study investigating time burden and costs in the UK.

Anita MacDonald, T Alexander Smith, Shamika de Silva, Veronica Alam, Jeanni M T van Loon

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Article in Molecular genetics and metabolism reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 35 papers, 3 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
35citing papers in PubMed, 3 pooled it
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1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

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3 · Its place in the literature

Who cites it

35 citing papers in PubMed, 3 syntheses or guidelines pooled it.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors.

Anita MacDonaldDietetic Department, The Children's Hospital, Birmingham, B4 6NH, UK.
T Alexander SmithMapi Ltd, London, N1 9BE, UK.
Shamika de SilvaMapi Ltd, London, N1 9BE, UK.
Veronica AlamEMD Serono Inc., Rockland, MA, USA.
Jeanni M T van LoonMapi Ltd, 3995 AX Houten, The Netherlands.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

introductionManagement of phenylketonuria (PKU) is mainly achieved through strict dietary control that aims to limit the intake of phenylalanine (Phe). Adherence to this diet is burdensome due to the need for specially prepared low-Phe meals and regular monitoring of Phe concentrations. A UK cross-sectional study was conducted to identify the personal time and monetary burden associated with aspects of the PKU lifestyle for caregivers of children (aged < 18 years) living with PKU.

methodsCaregivers of pediatric patients with PKU attending one of four specialist metabolic centers in the UK were invited to participate in a questionnaire-based survey that evaluated different aspects of PKU management that could potentially present out-of-pocket costs (OOPCs) or time burden. Medical clinicians/dieticians provided patient information on PKU severity and an assessment of blood Phe control.

resultsThe survey was completed by 114 caregivers of 106 children having mild or moderate (n = 45; 39%) or classical (n = 60; 53%) PKU (severity data missing for n = 1), among whom 8 (8%) and 87 (82%) reported poorly controlled and controlled blood Phe status, respectively; Phe control data were missing for 11 children. Dietary management of PKU incurred a median time burden of > 19 h per week. OOPCs were incurred via attendance at PKU events, PKU-related equipment, and extra holiday expenditure. 21% of caregivers reduced their working hours (median 18.5 h/week) to care for their child, with a further 24% leaving their paid jobs completely. DISCUSSION AND

conclusionsDietary management of PKU is associated with a considerable time burden for caregivers of pediatric patients with PKU. A personal financial burden also arises from OOPCs and lost earnings.

Indexed as

ACBS, Advisory Committee on Borderline SubstancesBurdenCaregiverDietDLA, Disability Living AllowanceNHS, National Health ServiceNRES, NHS Research Ethics CommitteesOOPC, out-of-pocket costPAH, phenylalanine hydroxylasePediatricPhenylalaninePhenylketonuriaPhe, phenylalaninePKU, phenylketonuria

Identifiers

PMID27622144
PMCPMC5009233

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.