Literature DB >> 26091594

Evaluation of Neuropsychiatric Function in Phenylketonuria: Psychometric Properties of the ADHD Rating Scale-IV and Adult ADHD Self-Report Scale Inattention Subscale in Phenylketonuria.

Kathleen W Wyrwich1, Priscilla Auguste2, Ren Yu2, Charlie Zhang3, Benjamin Dewees3, Barbara Winslow3, Shui Yu3, Markus Merilainen3, Suyash Prasad3.   

Abstract

BACKGROUND: Previous qualitative research among adults and parents of children with phenylketonuria (PKU) has identified inattention as an important psychiatric aspect of this condition. The parent-reported ADHD Rating Scale-IV (ADHD RS-IV) and the Adult ADHD Self-Report Scale (ASRS) have been validated for measuring inattention symptoms in persons with attention-deficit/hyperactivity disorder (ADHD); however, their psychometric attributes for measuring PKU-related inattention have not been established.
OBJECTIVE: The primary objective of this investigation was to demonstrate the reliability, validity, and responsiveness of the ADHD RS-IV and ASRS inattention symptoms subscales in a randomized controlled trial of patients with PKU aged 8 years or older.
METHODS: A post hoc analysis investigated the psychometric properties (Rasch model fit, reliability, construct validity, and responsiveness) of the ADHD RS-IV and ASRS inattention subscales using data from a phase 3b, double-blind, placebo-controlled clinical trial in those with PKU aged 8 years or older.
RESULTS: The Rasch results revealed good model fit, and reliability analyses revealed strong internal consistency reliability (α ≥ 0.87) and reproducibility (intraclass correlation coefficient ≥ 0.87) for both measures. Both inattention measures demonstrated the ability to discriminate between known groups (P < 0.001) created by the Clinical Global Impression-Severity scale. Correlations between the ADHD RS-IV and the ASRS with the Clinical Global Impression-Severity scale and the age-appropriate Behavior Rating Inventory of Executive Function Working Memory subscale were consistently moderate to strong (r ≥ 0.56). Similarly, results of the change score correlations were of moderate magnitude (r ≥ 0.43) for both measures when compared with changes over time in Behavior Rating Inventory of Executive Function Working Memory subscales.
CONCLUSIONS: These findings of reliability, validity, and responsiveness of both the ADHD RS-IV and the ASRS inattention scales, in addition to content validation results, support their use for the assessment of inattention symptoms among persons with PKU aged 8 years or older in both clinical and research settings.
Copyright © 2015 International Society for Pharmacoeconomics and Outcomes Research (ISPOR). Published by Elsevier Inc. All rights reserved.

Entities:  

Keywords:  attention deficit/hyperactivity disorder; inattention; metabolic disorder; phenylketonuria; psychometrics

Mesh:

Year:  2015        PMID: 26091594     DOI: 10.1016/j.jval.2015.01.008

Source DB:  PubMed          Journal:  Value Health        ISSN: 1098-3015            Impact factor:   5.725


  4 in total

1.  Improved attention linked to sustained phenylalanine reduction in adults with early-treated phenylketonuria.

Authors:  Deborah A Bilder; Georgianne L Arnold; David Dimmock; Mitzie L Grant; Darren Janzen; Nicola Longo; Mina Nguyen-Driver; Elaina Jurecki; Markus Merilainen; Gianni Amato; Susan Waisbren
Journal:  Am J Med Genet A       Date:  2021-11-26       Impact factor: 2.578

2.  Clinical and neuropsychological outcomes for children with phenylketonuria in Upper Egypt; a single-center study over 5 years.

Authors:  Abdelrahim A Sadek; Mohammed H Hassan; Nesreen A Mohammed
Journal:  Neuropsychiatr Dis Treat       Date:  2018-10-05       Impact factor: 2.570

3.  Behavioral and Emotional Problems in Early-Treated Brazilian Children and Adolescents with Phenylketonuria.

Authors:  Eduardo Vieira Neto; Heber de Souza Maia Filho; Claudia Braga Monteiro; Lilian de Mattos Carvalho; Thuane Silva da Cruz; Belisia Vasconcelos de Barros; Marcia Gonçalves Ribeiro
Journal:  Med Sci Monit       Date:  2018-10-30

4.  Long-term efficacy and safety of sapropterin in patients who initiated sapropterin at < 4 years of age with phenylketonuria: results of the 3-year extension of the SPARK open-label, multicentre, randomised phase IIIb trial.

Authors:  Ania C Muntau; Alberto Burlina; François Eyskens; Peter Freisinger; Vincenzo Leuzzi; Hatice Serap Sivri; Gwendolyn Gramer; Renata Pazdírková; Maureen Cleary; Amelia S Lotz-Havla; Paul Lane; Ignacio Alvarez; Frank Rutsch
Journal:  Orphanet J Rare Dis       Date:  2021-08-03       Impact factor: 4.123

  4 in total

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