{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,7]],"date-time":"2026-09-07T15:52:07Z","timestamp":1788796327786,"version":"build-2803163510"},"reference-count":52,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2024,2,16]],"date-time":"2024-02-16T00:00:00Z","timestamp":1708041600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Economic and Social Research Council (ESRC) and the National Institute for Health Research (NIHR)","award":["ES\/S010467\/1"],"award-info":[{"award-number":["ES\/S010467\/1"]}]},{"name":"Wales Centre of Ageing and Dementia Research (CADR)","award":["ES\/S010467\/1"],"award-info":[{"award-number":["ES\/S010467\/1"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJERPH"],"abstract":"<jats:p>The pathways for receiving a diagnosis of a rare type of dementia are poorly understood. Diagnostic challenges decrease access to relevant health promotion activities and post-diagnostic support. This study was focused on pathways experienced by people affected by rare dementia in Wales, United Kingdom (UK), considering the practical, emotional, and economic consequences. Semi-structured interviews were completed with 10 people affected by rare dementia across Wales, UK (nine family caregivers and one person living with rare dementia). The interview data were subject to a thematic analysis and a bottom-up costing approach was used to cost the pathway journeys. Five transitional points occurred across the diagnostic pathway (initial contact, initial referral, further referrals\u2014provider, further referrals\u2014private, and diagnosis) alongside two further themes (i.e., involved in the diagnostic process and disputes between stakeholders). The timeliness of the diagnosis was perceived to often be subject to \u2018luck\u2019, with access to private healthcare a personal finance option to expedite the process. Higher economic costs were observed when, in retrospect, inappropriate referrals were made, or multiple referrals were required. The confusion and disputes relating to individual diagnostic pathways led to further emotional burdens, suggesting that higher economic costs and emotional consequences are interlinked. Clearer diagnostic pathways for rare dementia may prevent unnecessary service contacts, waiting times, and associated distress. Prioritising appropriate and timely service contacts leads to diagnosis and support to families and enables people to increase control over their health. Appropriate diagnostic pathways may be less costly and reduce costs for families.<\/jats:p>","DOI":"10.3390\/ijerph21020231","type":"journal-article","created":{"date-parts":[[2024,2,16]],"date-time":"2024-02-16T03:37:27Z","timestamp":1708054647000},"page":"231","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["The Diagnostic Pathway Experiences of People Living with Rare Dementia and Their Family Caregivers: A Cross-Sectional Mixed Methods Study Using Qualitative and Economic Analyses"],"prefix":"10.3390","volume":"21","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4639-5338","authenticated-orcid":false,"given":"Ian","family":"Davies-Abbott","sequence":"first","affiliation":[{"name":"The Centre for Applied Dementia Studies, Faculty of Health Studies, University of Bradford, Bradford BD7 1DP, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bethany F.","family":"Anthony","sequence":"additional","affiliation":[{"name":"DSDC Wales Research Centre, School of Health Sciences, Bangor University, Bangor LL57 2PZ, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kiara","family":"Jackson","sequence":"additional","affiliation":[{"name":"DSDC Wales Research Centre, School of Health Sciences, Bangor University, Bangor LL57 2PZ, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gill","family":"Windle","sequence":"additional","affiliation":[{"name":"DSDC Wales Research Centre, School of Health Sciences, Bangor University, Bangor LL57 2PZ, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rhiannon Tudor","family":"Edwards","sequence":"additional","affiliation":[{"name":"DSDC Wales Research Centre, School of Health Sciences, Bangor University, Bangor LL57 2PZ, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,2,16]]},"reference":[{"key":"ref_1","unstructured":"Gauthier, S., Rosa-Neto, P., Morais, J.A., and Webster, C. (2021). World Alzheimer Report 2021: Journey though the Diagnosis of Dementia, Alzheimer Disease International. Available online: https:\/\/www.alzint.org\/u\/World-Alzheimer-Report-2021.pdf."},{"key":"ref_2","unstructured":"(2023, April 25). Alzheimer\u2019s Society. Available online: https:\/\/www.alzheimers.org.uk\/."},{"key":"ref_3","unstructured":"(2023, May 10). Alzheimer\u2019s Research UK (2020) Dementia Statistics Hub: Diagnoses in the UK. Available online: https:\/\/www.dementiastatistics.org\/statistics\/diagnoses-in-the-uk\/."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"384","DOI":"10.1192\/bjp.168.3.384b","article-title":"Non-Alzheimer dementias in young patients","volume":"168","author":"Harvey","year":"2003","journal-title":"Br. J. Psychiatry"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"785","DOI":"10.1016\/S1474-4422(11)70156-9","article-title":"Neuropathologically defined subtypes of Alzheimer\u2019s disease with distinct clinical characteristics: A retrospective study","volume":"10","author":"Murray","year":"2011","journal-title":"Lancet Neurol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"126","DOI":"10.4088\/JCP.10m06382oli","article-title":"The diagnostic challenge of psychiatric symptoms in neurodegenerative disease; rates of and risk factors for prior psychiatric diagnosis in patients with early neurodegenerative disease","volume":"72","author":"Woolley","year":"2011","journal-title":"J. Clin. Psychiatry"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"833","DOI":"10.1002\/gps.5253","article-title":"Protocol for the Rare Dementia Support Impact study: RDS Impact","volume":"35","author":"Brotherhood","year":"2020","journal-title":"Int. J. Geriatr. Psychiatry"},{"key":"ref_8","unstructured":"(2022, December 10). Rare Dementia Support. Available online: https:\/\/www.raredementiasupport.org\/."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"1080","DOI":"10.1001\/jamaneurol.2021.2161","article-title":"Global Prevalence of Young-Onset Dementia: A systematic review and meta-analysis","volume":"78","author":"Hendriks","year":"2021","journal-title":"JAMA Neurol."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"455","DOI":"10.1177\/1471301209349106","article-title":"Having a father with young onset dementia: The impact on well-being of young people","volume":"8","author":"Allen","year":"2009","journal-title":"Dementia"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1177\/1471301218783542","article-title":"\u201cI have to be both mother and father\u201d: The impact of Young-onset dementia on the partner\u2019s parenting and the children\u2019s experience","volume":"19","author":"Gelman","year":"2020","journal-title":"Dementia"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"356","DOI":"10.1017\/S1041610210001353","article-title":"The impact of young onset dementia on the family: A literature review","volume":"23","author":"Svanberg","year":"2011","journal-title":"Int. Psychogeriatr."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"1091","DOI":"10.1002\/gps.2439","article-title":"Impact of early onset dementia on caregivers: A review","volume":"25","author":"Bakker","year":"2010","journal-title":"Int. J. Geriatr. Psychiatry"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"2700","DOI":"10.1177\/14713012221126368","article-title":"Peer support for people living with rare or young onset dementia: An integrative review","volume":"21","author":"Sullivan","year":"2020","journal-title":"Dementia"},{"key":"ref_15","unstructured":"Department of Health (2023, January 23). Prime Minister\u2019s Challenge on Dementia 2020 Implementation Plan, Available online: https:\/\/assets.publishing.service.gov.uk\/government\/uploads\/system\/uploads\/attachment_data\/file\/507981\/PM_Dementia-main_acc.pdf."},{"key":"ref_16","unstructured":"Welsh Government (2023, January 23). Dementia Action Plan for Wales 2018\u20132022. Available online: https:\/\/gov.wales\/sites\/default\/files\/publications\/2019-04\/dementia-action-plan-for-wales.pdf."},{"key":"ref_17","unstructured":"National Institute for Health and Care Excellence (2023, February 04). Dementia\u2014Assessment, Management and Support for People Living with Dementia and Their Carers. Available online: https:\/\/www.nice.org.uk\/guidance\/indevelopment\/gid-cgwave0792\/consultation\/html-content-3."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"611","DOI":"10.1017\/S1041610219002229","article-title":"Persistent barriers and facilitators to seeking help for a dementia diagnosis: A systematic review of 30 years of the perspectives of carers and people with dementia","volume":"32","author":"Parker","year":"2020","journal-title":"Int. Psychogeriatr."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"514","DOI":"10.1177\/1471301216682625","article-title":"Identifying barriers to diagnosing dementia following incentivisation and policy pressures: General practitioners\u2019 perspectives","volume":"18","author":"Chithiramohan","year":"2019","journal-title":"Dementia"},{"key":"ref_20","first-page":"3637954","article-title":"Physician Perceptions about the Barriers to Prompt Diagnosis of Mild Cognitive Impairment and Alzheimer\u2019s Disease","volume":"2019","author":"Judge","year":"2019","journal-title":"Int. J. Alzheimer\u2019s Dis."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"3004","DOI":"10.1177\/1471301218763904","article-title":"\u2018How do they want to know?\u2019 Doctors\u2019 perspectives on making and communicating a diagnosis of dementia","volume":"18","author":"Bailey","year":"2018","journal-title":"Dementia"},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"1050","DOI":"10.1177\/14713012211067882","article-title":"Truth, hope and the disclosure of a dementia diagnosis: A scoping review of the ethical considerations from the perspective of the person, carer and clinician","volume":"21","author":"Merl","year":"2022","journal-title":"Dementia"},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1605","DOI":"10.1080\/13607863.2018.1506747","article-title":"Exploring the experience of the disclosure of a dementia diagnosis from a clinician, patient and carer perspective: A systematic review and Meta-ethnographic synthesis","volume":"23","author":"Poyser","year":"2018","journal-title":"Aging Ment. Health"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"e019060","DOI":"10.1136\/bmjopen-2017-019060","article-title":"Resource utilisation and costs in predementia and dementia: A systematic review protocol","volume":"8","author":"Landeiro","year":"2018","journal-title":"BMJ Open"},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1186\/s13195-017-0290-6","article-title":"Cost of diagnosing dementia in a German memory clinic","volume":"9","author":"Michalowsky","year":"2017","journal-title":"Alzheimer\u2019s Res. Ther."},{"key":"ref_26","unstructured":"Prince, M., Knapp, M., Guerchet, M., McCrone, P., Prina, M., Comas-Herrera, M., Wittenberg, A., Adelaja, R., Hu, B., and King, B. (2023, July 03). Dementia UK: Update. Alzheimer\u2019s Society 2014. Available online: http:\/\/www.alzheimers.org.uk\/dementiauk-."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"2041","DOI":"10.1017\/S0144686X21001495","article-title":"\u2018We\u2019re happy as we are\u2019: The experience of living with possible undiagnosed dementia","volume":"43","author":"Henley","year":"2021","journal-title":"Ageing Soc."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"2954","DOI":"10.1111\/jan.13031","article-title":"Systematic methodological review: Developing a framework for a qualitative semi-structured interview guide","volume":"72","author":"Kallio","year":"2016","journal-title":"J. Adv. Nurs."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"77","DOI":"10.1191\/1478088706qp063oa","article-title":"Using thematic analysis in psychology","volume":"3","author":"Braun","year":"2006","journal-title":"Qual. Res. Psychol."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"26152","DOI":"10.3402\/qhw.v9.26152","article-title":"What Can \u201cThematic Analysis\u201d Offer Health and Wellbeing Researchers?","volume":"9","author":"Braun","year":"2014","journal-title":"Int. J. Qual. Stud. Health Wellbeing"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1007\/s11121-012-0302-5","article-title":"Micro-Costing in Public Health Economics: Steps Towards a Standardized Framework, Using the Incredible Years Toddler Parenting Program as a Worked Example","volume":"14","author":"Charles","year":"2013","journal-title":"Prev. Sci."},{"key":"ref_32","unstructured":"Edwards, R.T., and McIntosh, E. (2019). Applied Health Economics for Public Health Practice and Research, Oxford University Press."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"479","DOI":"10.1017\/lst.2018.55","article-title":"Between \u2018going private\u2019 and \u2018NHS privatisation\u2019: Patient choice, competition reforms and the relationship between the NHS and private healthcare in England","volume":"39","author":"Guy","year":"2019","journal-title":"Leg. Stud."},{"key":"ref_34","unstructured":"UK Government (2023, June 22). National Minimum Wage and National Living Wage rates. UK Government, Available online: https:\/\/www.gov.uk\/national-minimum-wage-rates."},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"1428","DOI":"10.1016\/j.jval.2018.04.1828","article-title":"Time Is Money: Investigating the Value of Leisure Time and Unpaid Work","volume":"21","author":"Verbooy","year":"2018","journal-title":"Value Health"},{"key":"ref_36","unstructured":"(2024, February 06). Mental Capacity Act. Department of Health. London, HMSO, Available online: https:\/\/www.legislation.gov.ukukpga\/2005\/9\/contents\/enacted."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"2708","DOI":"10.1177\/14713012211009341","article-title":"Family carers\u2019 narratives of the financial consequences of young onset dementia","volume":"20","author":"Bayly","year":"2021","journal-title":"Dementia"},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1108\/JICA-02-2020-0005","article-title":"Mapping post-diagnostic dementia care in England: An e-survey","volume":"29","author":"Frost","year":"2021","journal-title":"J. Integr. Care"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"76","DOI":"10.1186\/s12939-021-01418-1","article-title":"\u201cA piece of paper is not the same as having someone to talk to\u201d: Accessing post-diagnostic dementia care before and since COVID-19 and associated inequalities","volume":"20","author":"Giebel","year":"2021","journal-title":"Int. J. Equity Health"},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"1125","DOI":"10.1080\/13607863.2020.1727854","article-title":"The nature of positive post-diagnostic support as experienced by people with young onset dementia","volume":"25","author":"Stamou","year":"2020","journal-title":"Aging Ment. Health"},{"key":"ref_41","first-page":"563","article-title":"How I Learned to Design and Conduct Semi-structured Interviews: An Ongoing and Continuous Journey","volume":"16","author":"Rabionet","year":"2011","journal-title":"Qual. Rep."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"056","DOI":"10.4103\/2321-0656.109828","article-title":"Three types of interviews: Qualitative research methods in social health","volume":"1","author":"Stuckey","year":"2013","journal-title":"J. Soc. Health Diabetes"},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"265","DOI":"10.1108\/17465641111188420","article-title":"The telephone medium and semi-structured interviews: A complementary fit","volume":"6","author":"Cachia","year":"2011","journal-title":"Qual. Res. Organ. Manag. Int. J."},{"key":"ref_44","unstructured":"Jones, K.C., and Burns, A. (2023, June 08). Unit costs of health and social care 2021 Personal Social Services Research Unit. Available online: https:\/\/www.pssru.ac.uk\/project-pages\/unit-costs\/unit-costs-of-health-and-social-care-2021\/."},{"key":"ref_45","unstructured":"(2023, November 01). National Schedule of NHS costs 2020\u201321. Available online: https:\/\/www.england.nhs.uk\/publication\/2020-21-national-cost-collection-data-publication\/."},{"key":"ref_46","unstructured":"(2023, November 01). NHS National Tariff Workbook 2020\/21 (Annex A). Available online: https:\/\/www.england.nhs.uk\/wp-content\/uploads\/2021\/02\/20-21NT_Annex_A_National_tariff_workbook.xlsx."},{"key":"ref_47","unstructured":"(2023, November 01). Information for patients who are having a CT scan. Available online: https:\/\/www.cuh.nhs.uk\/patient-information\/information-for-patients-who-are-having-a-ct-scan\/."},{"key":"ref_48","unstructured":"(2023, November 01). Nuffield Health Hospital Guide Price. Available online: https:\/\/www.nuffieldhealth.com\/consultants?size=n_20_n&sort-field=sortname&sort-direction=asc."},{"key":"ref_49","unstructured":"(2023, November 01). Specsavers. Available online: https:\/\/www.specsavers.co.uk\/help-and-faqs\/how-much-is-an-eye-test."},{"key":"ref_50","unstructured":"(2023, November 01). The College of Optometrists. Available online: https:\/\/lookafteryoureyes.org\/eye-examinations\/the-eye-examination\/#:~:text=The%20eye%20examination%20usually%20takes,your%20general%20health."},{"key":"ref_51","unstructured":"(2023, June 08). Prescription Cost Analysis. Available online: https:\/\/www.nhsbsa.nhs.uk\/statistical-collections\/prescription-cost-analysis-england\/prescription-cost-analysis-england-2020-21."},{"key":"ref_52","unstructured":"(2023, October 08). Travel\u2014Mileage and Fuel Rates and Allowances. HMRC, Available online: https:\/\/www.gov.uk\/government\/publications\/rates-and-allowances-travel-mileage-and-fuel-allowances\/travel-mileage-and-fuel-rates-and-allowances."}],"container-title":["International Journal of Environmental Research and Public Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1660-4601\/21\/2\/231\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T14:00:30Z","timestamp":1760104830000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1660-4601\/21\/2\/231"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,2,16]]},"references-count":52,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2024,2]]}},"alternative-id":["ijerph21020231"],"URL":"https:\/\/doi.org\/10.3390\/ijerph21020231","relation":{},"ISSN":["1660-4601"],"issn-type":[{"value":"1660-4601","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,2,16]]}}}