{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,11]],"date-time":"2026-10-11T14:16:06Z","timestamp":1791728166686,"version":"4.3.4"},"reference-count":28,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,5,25]],"date-time":"2026-05-25T00:00:00Z","timestamp":1779667200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"},{"start":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T00:00:00Z","timestamp":1784592000000},"content-version":"vor","delay-in-days":57,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Health Serv Res"],"DOI":"10.1186\/s12913-026-14801-x","type":"journal-article","created":{"date-parts":[[2026,5,25]],"date-time":"2026-05-25T10:24:59Z","timestamp":1779704699000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Out-of-pocket expenditures, accessibility, and affordability of low-protein nutrition in rare metabolic disorders in T\u00fcrkiye"],"prefix":"10.1186","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1286-903X","authenticated-orcid":false,"given":"M\u00fcjdat","family":"Ye\u015fildal","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2490-391X","authenticated-orcid":false,"given":"Mehmet Akif","family":"Eri\u015fen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4968-9828","authenticated-orcid":false,"given":"Merve","family":"E\u015fgi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,5,25]]},"reference":[{"issue":"1","key":"14801_CR1","first-page":"31","volume":"29","author":"RA Williams","year":"2008","unstructured":"Williams RA, Mamotte CDS, Burnett JR. Phenylketonuria: an inborn error of phenylalanine metabolism. Clin Biochem Rev. 2008;29(1):31\u201341.","journal-title":"Clin Biochem Rev"},{"key":"14801_CR2","unstructured":"Republic of Turkey Ministry of Health, General Directorate of Public Health. National screening program: phenylketonuria and other metabolic disorders statistics [Internet]. 2024. Available from: https:\/\/hsgm.saglik.gov.tr\/depo\/birimler\/cocuk-ergen-sagligi-db\/Dokumanlar\/Istatistikler\/NTP.pdf."},{"issue":"3","key":"14801_CR3","doi-asserted-by":"publisher","first-page":"139","DOI":"10.1016\/0925-6164(95)00125-5","volume":"4","author":"I \u00d6zalp","year":"1995","unstructured":"\u00d6zalp I, Co\u015fkun T, Tokatli A, Tokol S, \u00d6zg\u00fc\u00e7 M, K\u00f6ksal G, et al. Neonatal PKU screening in Turkey: 7 years experience in a developing country. Screening. 1995;4(3):139\u201347.","journal-title":"Screening"},{"issue":"2","key":"14801_CR4","doi-asserted-by":"publisher","first-page":"34","DOI":"10.3345\/kjp.2019.00465","volume":"63","author":"HR Shoraka","year":"2020","unstructured":"Shoraka HR, Haghdoost AA, Baneshi MR, Bagherinezhad Z, Zolala F. Global prevalence of classic phenylketonuria based on Neonatal Screening Program Data: systematic review and meta-analysis. Clin Exp Pediatr. 2020;63(2):34\u201343.","journal-title":"Clin Exp Pediatr"},{"issue":"1","key":"14801_CR5","doi-asserted-by":"publisher","first-page":"78","DOI":"10.1186\/s13023-024-03079-z","volume":"19","author":"M Esgi","year":"2024","unstructured":"Esgi M, Ergun H, Kaya NY, Atakay DY, Erucar E, Celik F. Phenylketonuria from the perspectives of patients in T\u00fcrkiye. Orphanet J Rare Dis. 2024;19(1):78.","journal-title":"Orphanet J Rare Dis"},{"issue":"7","key":"14801_CR6","first-page":"CD001304","volume":"7","author":"E Jameson","year":"2020","unstructured":"Jameson E, Remmington T. Dietary interventions for phenylketonuria. Cochrane Database Syst Rev. 2020;7(7):CD001304.","journal-title":"Cochrane Database Syst Rev"},{"issue":"6","key":"14801_CR7","doi-asserted-by":"publisher","first-page":"665","DOI":"10.1007\/s10545-010-9073-y","volume":"33","author":"A MacDonald","year":"2010","unstructured":"MacDonald A, Gokmen-Ozel H, van Rijn M, Burgard P. The reality of dietary compliance in the management of phenylketonuria. J Inherit Metab Dis. 2010;33(6):665\u201370.","journal-title":"J Inherit Metab Dis"},{"issue":"8","key":"14801_CR8","doi-asserted-by":"publisher","first-page":"628","DOI":"10.1080\/1028415X.2018.1538196","volume":"23","author":"PD Manta-Vogli","year":"2020","unstructured":"Manta-Vogli PD, Dotsikas Y, Loukas YL, Schulpis KH. The phenylketonuria patient: A recent dietetic therapeutic approach. Nutr Neurosci. 2020;23(8):628\u201339.","journal-title":"Nutr Neurosci"},{"key":"14801_CR9","doi-asserted-by":"publisher","first-page":"S19","DOI":"10.1016\/j.ymgme.2011.08.009","volume":"104","author":"A B\u00e9langer-Quintana","year":"2011","unstructured":"B\u00e9langer-Quintana A, Burlina A, Harding CO, Muntau AC. Up to date knowledge on different treatment strategies for phenylketonuria. Mol Genet Metab. 2011;104:S19\u201325.","journal-title":"Mol Genet Metab"},{"issue":"5","key":"14801_CR10","doi-asserted-by":"publisher","first-page":"639","DOI":"10.1007\/s10545-005-4478-8","volume":"28","author":"C Bilginsoy","year":"2005","unstructured":"Bilginsoy C, Waitzman N, Leonard CO, Ernst SL. Living with phenylketonuria: perspectives of patients and their families. J Inherit Metab Dis. 2005;28(5):639\u201349.","journal-title":"J Inherit Metab Dis"},{"issue":"5","key":"14801_CR11","doi-asserted-by":"publisher","first-page":"193","DOI":"10.1007\/s11011-025-01582-1","volume":"40","author":"FN Kuzucu","year":"2025","unstructured":"Kuzucu FN, Kilic M, Sezer A, Has-Ozhan S, Yildiz H, Celen-Yoldas T, et al. Genotypic and phenotypic characteristics of Turkish patients with phenylalanine metabolism disorders. Metab Brain Dis. 2025;40(5):193.","journal-title":"Metab Brain Dis"},{"issue":"5","key":"14801_CR12","doi-asserted-by":"publisher","first-page":"335","DOI":"10.1111\/j.1600-0404.2009.01184.x","volume":"120","author":"R Lv","year":"2009","unstructured":"Lv R, Wu L, Jin L, Lu Q, Wang M, Qu Y, et al. Depression, anxiety and quality of life in parents of children with epilepsy. Acta Neurol Scand. 2009;120(5):335\u201341.","journal-title":"Acta Neurol Scand"},{"issue":"6","key":"14801_CR13","doi-asserted-by":"publisher","first-page":"673","DOI":"10.1007\/s10545-008-0918-6","volume":"31","author":"FJ van Spronsen","year":"2008","unstructured":"van Spronsen FJ, Burgard P. The truth of treating patients with phenylketonuria after childhood: the need for a new guideline. J Inherit Metab Dis. 2008;31(6):673\u20139.","journal-title":"J Inherit Metab Dis"},{"issue":"3","key":"14801_CR14","doi-asserted-by":"publisher","first-page":"392","DOI":"10.3390\/nu17030392","volume":"17","author":"S Evans","year":"2025","unstructured":"Evans S, Arbuckle C, Ashmore C, Bailey S, Blaauw G, Chaudhry W, et al. UK Patient Access to Low-Protein Prescription Foods in Phenylketonuria (PKU): An Uneasy Path. Nutrients. 2025;17(3):392.","journal-title":"Nutrients"},{"key":"14801_CR15","first-page":"101080","volume":"39","author":"A Andrews","year":"2024","unstructured":"Andrews A, McMinimee K. Navigating social determinants of health barriers in the management of phenylketonuria. Mol Genet Metabolism Rep. 2024;39:101080.","journal-title":"Mol Genet Metabolism Rep"},{"key":"14801_CR16","doi-asserted-by":"publisher","first-page":"43","DOI":"10.1186\/1472-6963-7-43","volume":"7","author":"N Devadasan","year":"2007","unstructured":"Devadasan N, Criel B, Van Damme W, Ranson K, Van der Stuyft P. Indian community health insurance schemes provide partial protection against catastrophic health expenditure. BMC Health Serv Res. 2007;7:43.","journal-title":"BMC Health Serv Res"},{"issue":"8","key":"14801_CR17","first-page":"612","volume":"80","author":"K Kawabata","year":"2002","unstructured":"Kawabata K, Xu K, Carrin G. Preventing impoverishment through protection against catastrophic health expenditure. Bull World Health Organ. 2002;80(8):612.","journal-title":"Bull World Health Organ"},{"issue":"3","key":"14801_CR18","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1016\/j.ymgme.2013.05.003","volume":"109","author":"I Eijgelshoven","year":"2013","unstructured":"Eijgelshoven I, Demirdas S, Smith TA, van Loon JMT, Latour S, Bosch AM. The time consuming nature of phenylketonuria: a cross-sectional study investigating time burden and costs of phenylketonuria in the Netherlands. Mol Genet Metab. 2013;109(3):237\u201342.","journal-title":"Mol Genet Metab"},{"issue":"3","key":"14801_CR19","doi-asserted-by":"publisher","first-page":"369","DOI":"10.1007\/s10545-016-9995-0","volume":"40","author":"L Wang","year":"2017","unstructured":"Wang L, Zou H, Ye F, Wang K, Li X, Chen Z, et al. Household financial burden of phenylketonuria and its impact on treatment in China: a cross-sectional study. J Inherit Metab Dis. 2017;40(3):369\u201376.","journal-title":"J Inherit Metab Dis"},{"key":"14801_CR20","unstructured":"Tatar M, Mollahalilo\u011flu S, \u015eahin B, Ayd\u0131n S, Maresso A, Hern\u00e1ndez-Quevedo C. Turkey: health system review. Health Systems in Transition [Internet]. 2011 [cited 2025 Jul 9];13(6). Available from: https:\/\/iris.who.int\/handle\/10665\/330325."},{"key":"14801_CR21","first-page":"1787","volume":"14","author":"D A\u00e7\u0131k","year":"2024","unstructured":"A\u00e7\u0131k D, Alper Y. T\u00fcrk genel sa\u011fl\u0131k sigortas\u0131 kapsam\u0131nda sunulan sa\u011fl\u0131k hizmetlerinin sosyal koruma tabanlar\u0131 yakla\u015f\u0131m\u0131na g\u00f6re finansman bak\u0131m\u0131ndan de\u011ferlendirilmesi. OD\u00dcSOB\u0130AD. 2024;14:1787\u201398. 23. \u00c7al\u0131\u015fma Ekonomisi ve End\u00fcstri \u0130li\u015fkileri B\u00f6l\u00fcm Kongresi \u00d6zel Say\u0131s\u0131).","journal-title":"OD\u00dcSOB\u0130AD"},{"issue":"9886","key":"14801_CR22","doi-asserted-by":"publisher","first-page":"65","DOI":"10.1016\/S0140-6736(13)61051-X","volume":"382","author":"R Atun","year":"2013","unstructured":"Atun R, Ayd\u0131n S, Chakraborty S, S\u00fcmer S, Aran M, G\u00fcrol I, et al. Universal health coverage in Turkey: enhancement of equity. Lancet. 2013;382(9886):65\u201399.","journal-title":"Lancet"},{"key":"14801_CR23","doi-asserted-by":"crossref","unstructured":"G\u00fcnd\u00fcz M, Y\u00fcksel G\u00fcdek Y, Kasapkara \u00c7S. Out-of-pocket health expenditures in patients living with \u0131nborn errors of metabolism. Orphanet J Rare Dis [Internet]. 2023 Jul 6 [cited 2025 Jul 14];18(1). Available from: https:\/\/ojrd.biomedcentral.com\/articles\/10.1186\/s13023-023-02775-6.","DOI":"10.1186\/s13023-023-02775-6"},{"key":"14801_CR24","doi-asserted-by":"publisher","first-page":"1051851","DOI":"10.3389\/fpubh.2023.1051851","volume":"11","author":"G Ko\u00e7kaya","year":"2023","unstructured":"Ko\u00e7kaya G, Oguzhan G, \u00d6k\u00e7\u00fcn S, Kurnaz M. Out-of-pocket healthcare expenditures of Turkish households living with rare diseases. Front Public Health. 2023;11:1051851.","journal-title":"Front Public Health"},{"issue":"10","key":"14801_CR25","doi-asserted-by":"publisher","first-page":"1444","DOI":"10.3390\/nu16101444","volume":"16","author":"M Karaca Sahin","year":"2024","unstructured":"Karaca Sahin M, Aktuglu Zeybek AC, Zubarioglu T, Cansever MS, K\u0131yk\u0131m E. Comparison of Cost Analysis in Patients with Tetrahydrobiopterin-Responsive and Non-Responsive Phenylketonuria in Turkey. Nutrients. 2024;16(10):1444.","journal-title":"Nutrients"},{"key":"14801_CR26","unstructured":"Central Bank of the Republic of Turkey. Indicative exchange rates announced by the Central Bank of the Republic of Turkey at 15:30 on 15.03.2024 [Internet]. 2024. Available from: https:\/\/www.tcmb.gov.tr\/kurlar\/202403\/15032024.xml."},{"key":"14801_CR27","unstructured":"Republic of T\u00fcrkiye Ministry of Labour and Social Security. 2024 Y\u0131l\u0131nda Uygulanacak Yeni Asgari \u00dccret 17 Bin 2 TL Olarak Belirlendi [Internet]. December 27, 2024. [cited 2026 Feb 17]. Available from: https:\/\/www.csgb.gov.tr\/haberler\/27-12-2023\/."},{"key":"14801_CR28","first-page":"100523","volume":"21","author":"AM Rose","year":"2019","unstructured":"Rose AM, Grosse SD, Garcia SP, Bach J, Kleyn M, Simon NJE, et al. The financial and time burden associated with phenylketonuria treatment in the United States. Mol Genet Metabolism Rep. 2019;21:100523.","journal-title":"Mol Genet Metabolism Rep"}],"container-title":["BMC Health Services Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12913-026-14801-x","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12913-026-14801-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12913-026-14801-x.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T22:45:51Z","timestamp":1784587551000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s12913-026-14801-x"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,25]]},"references-count":28,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["14801"],"URL":"https:\/\/doi.org\/10.1186\/s12913-026-14801-x","relation":{},"ISSN":["1472-6963"],"issn-type":[{"value":"1472-6963","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,25]]},"assertion":[{"value":"9 April 2025","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"20 May 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"25 May 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The study was approved by the Tokat Gaziosmanpa\u015fa University Clinical Research Ethics Committee (Decision No: 23-KAEK-04, Date: 16.02.2023, Meeting No: 2023\/03). The study was conducted in accordance with the principles of the Declaration of Helsinki. Informed consent was obtained from all individual participants included in the study. For participants under 18, permission was obtained from legal guardians.","order":1,"name":"Ethics","label":"Ethics approval and consent to participate","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","label":"Consent for publication","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declare no competing interests.","order":3,"name":"Ethics","label":"Competing interests","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"1005"}}