Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers

Introduction: Rare kidney diseases constitute a significant public health challenge but have attracted limited research investment. The evidence about the socioeconomic burden of rare kidney diseases has not been systematically examined. Such evidence is critical for generating the advocacy and awar...

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Main Authors: Anna Palagyi, Agnivo Sengupta, Monica Moorthy, Charu Malik, Jonathan Barratt, Olivier Devuyst, Ifeoma I. Ulasi, Daniel P. Gale, Siyuan Wang, Blake Angell, Vivekanand Jha, Stephen Jan
Format: Article
Language:English
Published: Elsevier 2025-03-01
Series:Kidney International Reports
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Online Access:http://www.sciencedirect.com/science/article/pii/S2468024924034041
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author Anna Palagyi
Agnivo Sengupta
Monica Moorthy
Charu Malik
Jonathan Barratt
Olivier Devuyst
Ifeoma I. Ulasi
Daniel P. Gale
Siyuan Wang
Blake Angell
Vivekanand Jha
Stephen Jan
author_facet Anna Palagyi
Agnivo Sengupta
Monica Moorthy
Charu Malik
Jonathan Barratt
Olivier Devuyst
Ifeoma I. Ulasi
Daniel P. Gale
Siyuan Wang
Blake Angell
Vivekanand Jha
Stephen Jan
author_sort Anna Palagyi
collection DOAJ
description Introduction: Rare kidney diseases constitute a significant public health challenge but have attracted limited research investment. The evidence about the socioeconomic burden of rare kidney diseases has not been systematically examined. Such evidence is critical for generating the advocacy and awareness necessary to impel scientific and policy investment in targeted care in health systems worldwide. We aimed to evaluate the socioeconomic burden borne by patients with rare kidney diseases, their families, and caregivers, and the related psychosocial impact. Methods: We undertook a systemic scoping review of the recent evidence of the socioeconomic and psychosocial burden of rare kidney diseases, to identify gaps in the understanding of this burden across contexts and factors influencing them. Three databases and the grey literature were searched for relevant studies published in the 10 years before April 30, 2023. Results: Fifty-three articles met the inclusion criteria; one-quarter of these articles included rare disease cohorts in which the kidney was the primary organ affected, and 91% of studies were conducted in high-income countries. Evidence of substantial life-long socioeconomic burden emerged across the following 4 main categories: education (n = 17 articles [32%]), work and employment (n = 40 [75%] articles), psychosocial and emotional impact (n = 17 [32%]), and out-of-pocket expenses (n = 15 [28%]). Conclusion: Significant gaps in our understanding of the socioeconomic burden remain, particularly in lower-resource health systems, among traditionally marginalized populations, and for rare diseases for which kidney is the primary affected organ. Further exploration of socioeconomic burden within these populations is vital to inform effectively targeted investment in advocacy and health care innovation for affected individuals.
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spelling doaj-art-ce762eaddb914f5aa9d9b5c4c39ac5af2025-08-20T03:21:18ZengElsevierKidney International Reports2468-02492025-03-0110383885410.1016/j.ekir.2024.12.005Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their CaregiversAnna Palagyi0Agnivo Sengupta1Monica Moorthy2Charu Malik3Jonathan Barratt4Olivier Devuyst5Ifeoma I. Ulasi6Daniel P. Gale7Siyuan Wang8Blake Angell9Vivekanand Jha10Stephen Jan11The George Institute for Global Health, University of New South Wales, Sydney, New South Wales, Australia; Correspondence: Anna Palagyi, The George Institute for Global Health, PO Box M201, Missenden Rd, Sydney, New South Wales 2050, Australia.The George Institute for Global Health, University of New South Wales, Sydney, New South Wales, AustraliaInternational Society of Nephrology, Brussels, BelgiumInternational Society of Nephrology, Brussels, BelgiumDepartment of Cardiovascular Sciences, University of Leicester, Leicester, UKDepartment of Physiology, Mechanisms of Inherited Kidney Disorders, University of Zurich, Zurich, Switzerland; Division of Nephrology, Cliniques Universitaires Saint-Luc, UCLouvain, Brussels, BelgiumRenal Unit, Department of Medicine, College of Medicine, University of Nigeria, Ituku-Ozalla, Enugu, Nigeria; Renal Unit, Department of Medicine, University of Nigeria Teaching Hospital, Ituku-Ozalla, Enugu, Nigeria; Renal Unit, Department of Internal Medicine, Alex Ekwueme Federal University Teaching Hospital, Abakaliki, NigeriaNational Registry of Rare Kidney Diseases, Bristol, UK; Department of Renal Medicine, University College London, London, UKThe George Institute for Global Health, University of New South Wales, Sydney, New South Wales, AustraliaThe George Institute for Global Health, University of New South Wales, Sydney, New South Wales, AustraliaInternational Society of Nephrology, Brussels, Belgium; The George Institute for Global Health, University of New South Wales, New Delhi, India; School of Public Health, Imperial College London, London, UK; Prasanna School of Public Health, Manipal Academy of Higher Education, Manipal, Karnataka, IndiaThe George Institute for Global Health, University of New South Wales, Sydney, New South Wales, AustraliaIntroduction: Rare kidney diseases constitute a significant public health challenge but have attracted limited research investment. The evidence about the socioeconomic burden of rare kidney diseases has not been systematically examined. Such evidence is critical for generating the advocacy and awareness necessary to impel scientific and policy investment in targeted care in health systems worldwide. We aimed to evaluate the socioeconomic burden borne by patients with rare kidney diseases, their families, and caregivers, and the related psychosocial impact. Methods: We undertook a systemic scoping review of the recent evidence of the socioeconomic and psychosocial burden of rare kidney diseases, to identify gaps in the understanding of this burden across contexts and factors influencing them. Three databases and the grey literature were searched for relevant studies published in the 10 years before April 30, 2023. Results: Fifty-three articles met the inclusion criteria; one-quarter of these articles included rare disease cohorts in which the kidney was the primary organ affected, and 91% of studies were conducted in high-income countries. Evidence of substantial life-long socioeconomic burden emerged across the following 4 main categories: education (n = 17 articles [32%]), work and employment (n = 40 [75%] articles), psychosocial and emotional impact (n = 17 [32%]), and out-of-pocket expenses (n = 15 [28%]). Conclusion: Significant gaps in our understanding of the socioeconomic burden remain, particularly in lower-resource health systems, among traditionally marginalized populations, and for rare diseases for which kidney is the primary affected organ. Further exploration of socioeconomic burden within these populations is vital to inform effectively targeted investment in advocacy and health care innovation for affected individuals.http://www.sciencedirect.com/science/article/pii/S2468024924034041educationlost productivityrare kidney diseasesscoping reviewsocioeconomic burden
spellingShingle Anna Palagyi
Agnivo Sengupta
Monica Moorthy
Charu Malik
Jonathan Barratt
Olivier Devuyst
Ifeoma I. Ulasi
Daniel P. Gale
Siyuan Wang
Blake Angell
Vivekanand Jha
Stephen Jan
Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
Kidney International Reports
education
lost productivity
rare kidney diseases
scoping review
socioeconomic burden
title Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
title_full Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
title_fullStr Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
title_full_unstemmed Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
title_short Systematic Scoping Review of Socioeconomic Burden and Associated Psychosocial Impact in Patients With Rare Kidney Diseases and Their Caregivers
title_sort systematic scoping review of socioeconomic burden and associated psychosocial impact in patients with rare kidney diseases and their caregivers
topic education
lost productivity
rare kidney diseases
scoping review
socioeconomic burden
url http://www.sciencedirect.com/science/article/pii/S2468024924034041
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