Monitoring and integrated care coordination of patients with alpha-mannosidosis: A global Delphi consensus study

Kód výsledku v IS VaVaI

<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00064165%3A_____%2F24%3A10483347" target="_blank" >RIV/00064165:_____/24:10483347 - isvavai.cz</a>

Nalezeny alternativní kódy

RIV/00216208:11110/24:10483347

Výsledek na webu

<a href="https://verso.is.cuni.cz/pub/verso.fpl?fname=obd_publikace_handle&handle=xsp.WVsXFd" target="_blank" >https://verso.is.cuni.cz/pub/verso.fpl?fname=obd_publikace_handle&handle=xsp.WVsXFd</a>

DOI - Digital Object Identifier

<a href="http://dx.doi.org/10.1016/j.ymgme.2024.108519" target="_blank" >10.1016/j.ymgme.2024.108519</a>

Jazyk výsledku

angličtina

Název v původním jazyce

Monitoring and integrated care coordination of patients with alpha-mannosidosis: A global Delphi consensus study

Popis výsledku v původním jazyce

Introduction: Current literature lacks consensus on initial assessments and routine follow-up care of patients with alpha-mannosidosis (AM). A Delphi panel was conducted to generate and validate recommendations on best practices for initial assessment, routine follow-up care, and integrated care coordination of patients with AM. Methods: A modified Delphi method involving 3 rounds of online surveys was used. An independent administrator and 2 nonvoting physician co-chairs managed survey development, anonymous data collection, and analysis. A multidisciplinary panel comprising 20 physicians from 12 countries responded to 57 open-ended questions in the first survey. Round 2 consisted of 11 ranking questions and 44 voting statements. In round 3, panelists voted to validate 60 consensus statements. The panel response rate was &gt;= 95% in all 3 rounds. Panelists used 5-point Likert scales to indicate importance (score of &gt;3) or agreement (score of &gt;4). Consensus was defined a priori as &gt;= 75% agreement with &gt;= 75% of panelists voting. Results: Consensus was reached on 60 statements, encompassing 3 key areas: initial assessments, routine followup care, and treatment-related follow-up. The panel agreed on the type and frequency of assessments related to genetic testing, baseline evaluations, quality of life, biochemical measures, affected body systems, treatment received, and integrated care coordination in patients with AM. Forty-nine statements reached 90% to 100% consensus, 8 statements reached 80% to 85% consensus, and 1 statement reached 75% consensus. Two statements each reached consensus on 15 baseline assessments to be conducted at the initial follow-up visit after diagnosis in pediatric and adult patients. Conclusion: This is the first Delphi study providing internationally applicable, best-practice recommendations for monitoring patients with AM that may improve their care and well-being.

Název v anglickém jazyce

Monitoring and integrated care coordination of patients with alpha-mannosidosis: A global Delphi consensus study

Popis výsledku anglicky

Introduction: Current literature lacks consensus on initial assessments and routine follow-up care of patients with alpha-mannosidosis (AM). A Delphi panel was conducted to generate and validate recommendations on best practices for initial assessment, routine follow-up care, and integrated care coordination of patients with AM. Methods: A modified Delphi method involving 3 rounds of online surveys was used. An independent administrator and 2 nonvoting physician co-chairs managed survey development, anonymous data collection, and analysis. A multidisciplinary panel comprising 20 physicians from 12 countries responded to 57 open-ended questions in the first survey. Round 2 consisted of 11 ranking questions and 44 voting statements. In round 3, panelists voted to validate 60 consensus statements. The panel response rate was &gt;= 95% in all 3 rounds. Panelists used 5-point Likert scales to indicate importance (score of &gt;3) or agreement (score of &gt;4). Consensus was defined a priori as &gt;= 75% agreement with &gt;= 75% of panelists voting. Results: Consensus was reached on 60 statements, encompassing 3 key areas: initial assessments, routine followup care, and treatment-related follow-up. The panel agreed on the type and frequency of assessments related to genetic testing, baseline evaluations, quality of life, biochemical measures, affected body systems, treatment received, and integrated care coordination in patients with AM. Forty-nine statements reached 90% to 100% consensus, 8 statements reached 80% to 85% consensus, and 1 statement reached 75% consensus. Two statements each reached consensus on 15 baseline assessments to be conducted at the initial follow-up visit after diagnosis in pediatric and adult patients. Conclusion: This is the first Delphi study providing internationally applicable, best-practice recommendations for monitoring patients with AM that may improve their care and well-being.

Druh

J_imp - Článek v periodiku v databázi Web of Science

CEP obor

—

OECD FORD obor

30209 - Paediatrics

Projekt

—

Návaznosti

N - Vyzkumna aktivita podporovana z neverejnych zdroju

Rok uplatnění

2024

Kód důvěrnosti údajů

S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Název periodika

Molecular Genetics and Metabolism

ISSN

1096-7192

e-ISSN

1096-7206

Svazek periodika

142

Číslo periodika v rámci svazku

4

Stát vydavatele periodika

US - Spojené státy americké

Počet stran výsledku

14

Strana od-do

108519

Kód UT WoS článku

001278795500001

EID výsledku v databázi Scopus

2-s2.0-85198607494