Long-term impact of molar-incisor hypomineralisation and its management on children and their families: an 8-year follow-up study with mixed methods analysis

Long-term impact of molar-incisor hypomineralisation and its management on children and their families: an 8-year follow-up study with mixed methods analysis | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Long-term impact of molar-incisor hypomineralisation and its management on children and their families: an 8-year follow-up study with mixed methods analysis Mohammad A Kh Hussein This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-8200677/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Purpose To investigate long-term changes in oral health-related quality of life (OHRQoL) and dental fear/anxiety (DFA) following specialist management of molar-incisor hypomineralisation (MIH), assess family impact, and explore patient/parent experiences through qualitative analysis. Methods This prospective follow-up study re-recruited participants from a 2015–2017 baseline cohort of children with MIH referred to a UK specialist centre. Participants completed the same validated instruments used at baseline: Child Oral Health Impact Profile-Short Form 19 (COHIP-SF19) and Modified Child Dental Anxiety Scale-faces version (MCDASf). Additionally, parents completed the Family Impact Scale (FIS). Clinical records were reviewed retrospectively. Qualitative content analysis was performed on open-ended responses about treatment experiences. Changes were analysed using descriptive statistics and thematic analysis. Results Eighteen participants (22% re-recruitment rate; mean age 15.6 years; 13 males) were followed up 7–8 years post-baseline. At baseline, 11 had severe, 4 moderate, and 3 mild MIH. Treatment modalities included local anaesthesia (n = 9), general anaesthesia (n = 8), and inhalation sedation (n = 2). Mean COHIP-SF19 scores showed minimal overall change (baseline: 56.3, follow-up: 53.6), with functional well-being showing greatest improvement. MCDASf scores remained stable (baseline: 20.1, follow-up: 20.5). However, individual variation was substantial, with OHRQoL and DFA changes inversely correlated in 14/18 participants. Permanent molar extractions under local anaesthesia were associated with the largest DFA increases. Family impact was generally low (mean FIS: 6/56) but increased with journey time to the specialist centre (> 60 minutes). Qualitative analysis revealed two main themes: positive communication experiences and system challenges (waiting times, appointment availability). Conclusions This long-term follow-up study demonstrates heterogeneous outcomes following MIH management. While group means suggest stability, individual trajectories varied considerably. The inverse relationship between OHRQoL and DFA changes, the potential anxiety impact of extractions under local anaesthesia, and the burden of travel distance represent novel findings requiring further investigation. The mixed-methods approach provided valuable insights into patient/family experiences that quantitative measures alone would miss. MIH OHRQoL DFA FIS Long-term follow-up Mixed methods Figures Figure 1 Introduction Molar-incisor hypomineralisation (MIH) affects approximately 13–14% of children globally (Dave and Taylor 2018; Schwendicke et al. 2018; Lopes et al. 2021), presenting significant clinical challenges including hypersensitivity, post-eruptive breakdown, and aesthetic concerns (Linner et al. 2021; Rodd et al. 2007). While immediate treatment outcomes have been documented (Fragelli et al. 2015; Bekes et al. 2021), long-term impacts on affected children and their families remain poorly understood. The European Academy of Paediatric Dentistry's 2022 clinical guidelines specifically identified the need for longitudinal research examining psychosocial and economic aspects of MIH (Lygidakis et al. 2022). Previous cross-sectional studies have reported conflicting findings regarding MIH's impact on oral health-related quality of life (OHRQoL), with some demonstrating significant negative effects (Dantas-Neta et al. 2016; Elhennawy et al. 2022) while others found minimal impact (Dias et al. 2021). Similarly, evidence regarding dental fear and anxiety (DFA) in MIH-affected children remains inconsistent (Jälevik and Klingberg 2012; Kosma et al. 2016). Critically, no previous studies have prospectively tracked the same cohort from pre-treatment through long-term post-treatment follow-up using validated instruments, nor have they incorporated qualitative analysis of patient and family experiences. This study addresses these gaps by presenting 7–8 year follow-up data from a prospectively recruited cohort, combining quantitative assessment of OHRQoL and DFA changes with qualitative exploration of treatment experiences. We aimed to: (1) evaluate long-term changes in OHRQoL and DFA following specialist MIH management; (2) assess the impact on families; (3) identify factors associated with outcomes; and (4) explore patient/parent perspectives through qualitative analysis. Materials and Methods Study Design and Ethical Approval This prospective follow-up study investigated a cohort originally recruited during 2015-2017 at Leeds Dental Institute, UK. Ethical approval was obtained from NHS Research Ethics Committee Yorkshire and The Humber (REC: 21/PR/1069) and Leeds Teaching Hospitals NHS Trust (R&I: DT22/148475). Participants From 82 children with MIH in the original cohort (Al-Bahar 2017), all were invited to participate via postal invitation followed by telephone contact. The COVID-19 pandemic necessitated remote data collection methods. Inclusion criteria were: previous participation in the baseline study, confirmed MIH diagnosis, and consent/assent from both participant and parent/guardian. Data Collection Quantitative Measures Participants completed the same validated instruments used at baseline: COHIP-SF19 (Broder et al. 2012): 19-item scale assessing OHRQoL across three domains (oral health, functional well-being, socio-emotional well-being). Scores range 0-76, with higher scores indicating better OHRQoL. MCDASf (Howard and Freeman 2007): 8-item scale measuring dental anxiety using facial expressions. Scores range 8-40, with higher scores indicating greater anxiety. Parents completed: Family Impact Scale (FIS) (Locker et al. 2002): 14-item scale assessing impact across four domains (parental activities, parental emotions, family conflict, financial burden). Scores range 0-56, with higher scores indicating greater impact. Transportation questionnaire: mode, duration, cost, and perceived difficulty of journeys to the specialist centre. Clinical Data Electronic and physical paper patient records were systematically reviewed to extract: MIH severity at baseline (mild/moderate/severe per Da Costa-Silva et al. 2011 criteria), treatment modalities (local anaesthesia/inhalation sedation/general anaesthesia), procedures performed, and number of visits. Qualitative Data Open-ended questions explored experiences of care: Participants: difficulties encountered, positive aspects, suggestions for improvement Parents: what was done well, areas for improvement, additional comments Data Analysis Quantitative Analysis Descriptive statistics were calculated for all measures. Individual change scores were computed (follow-up minus baseline). Relationships between variables were explored graphically due to small sample size. Data were analysed using Microsoft Excel. Qualitative Analysis Content analysis followed established methodology (Krippendorff 2018). Two researchers independently reviewed responses, identifying recurring themes and developing codes. NVivo 1.7.1 facilitated systematic coding. Themes were refined through discussion until consensus was reached. Results Participant Characteristics Eighteen participants were recruited (22% response rate): 13 males, 5 females; mean age 15.6 years (range 14-17) at follow-up; mean 7.5 years since baseline. At baseline: 11 had severe MIH, 4 moderate, 3 mild. Treatment modalities included: local anaesthesia only (n=5), general anaesthesia only (n=6), combined approaches (n=3), no active intervention (n=4). Eleven participants underwent extractions, 7 received restorations. Quantitative Outcomes OHRQoL Changes Overall COHIP-SF19 scores showed minimal mean change (baseline: 56.3±9.5, follow-up: 53.6±12.3, mean change: -2.6). However, individual changes ranged from +18 to -23 points. Domain analysis revealed: Functional well-being : 12/18 improved (mean change: +1.0) Oral health : 7/18 improved (mean change: -0.4) Socio-emotional well-being : 8/18 improved (mean change: -3.1) Notably, all three mild MIH cases showed OHRQoL decline, while 7/11 severe cases improved. DFA Changes MCDASf scores remained stable overall (baseline: 20.1±6.8, follow-up: 20.5±7.4, mean change: +0.5), but individual changes ranged from -12 to +24. The two largest anxiety increases occurred in participants receiving permanent molar extractions under local anaesthesia (+24 and +10 points). Relationship Between OHRQoL and DFA Changes in COHIP-SF19 and MCDASf were inversely correlated in 14/18 participants: improved OHRQoL accompanied reduced anxiety or vice versa. Table1 Differences in COHIP-SF19 total scores and in MCDASf total scores, and their changes between baseline and follow-up studies, ranked according to the difference in total COHIP-SF19. Participant ID Difference in COHIP-SF19 total score Difference in MCDASf total score 108 18 -10 11 11 -3 28 6 -4 46 6 24 65 6 3 43 4 -2 66 4 -3 63 3 -8 12 2 -3 97 2 -12 30 -4 -2 51 -9 -5 107 -9 10 49 -11 5 99 -11 3 100 -19 8 2 -23 4 60 -23 3 Note - A positive change (increase) in total COHIP SF-19 is indicative of an improvement in OH QoL (indicated in green). A decline in COHIP-SF19 score is indicative of a decrease in OH QoL (Red). - A positive change (increase in MCDASf score indicates an increase in dental anxiety (indicated in Red). A decrease in MCDASf score is indicative of a decrease in dental anxiety (Green). Family Impact Mean FIS score was 6.0±6.7 (range 0-25), indicating generally low impact. All families reported zero financial burden. Impact increased with journey time: families travelling >60 minutes had mean FIS 13.7 versus 3.8 for <60 minutes. Parental activities (mean 2.8) and emotions (mean 2.0) were most affected domains. Qualitative Findings Two main themes emerged from content analysis: Theme 1: Communication Excellence Parents and participants consistently praised communication: "Everything was explained in detail to myself and my child" (Parent-028) "Great explanation of what was going on. Made [child] feel really comfortable" (Parent-063) "Staff were friendly" (Multiple respondents) Theme 2: System Challenges Waiting times and appointment availability were primary concerns: "Waited long time for appointments. Follow-up slow. Had to ring to chase appointments" (Parent-030) "The availability of appointments is quite limiting. I feel like treatment has taken longer due to this" (Parent-063) "Reduce waiting time as this increases anxiety" (Parent-108) Despite challenges, 17/18 families reported satisfaction with overall care. While one family found the experience to be neutral. Discussion This study presents the first long-term prospective follow-up of MIH-affected children using validated pre- and post-treatment measures combined with qualitative analysis, addressing a critical gap identified by EAPD guidelines (Lygidakis et al. 2022). Individual Variability in Outcomes While group means suggested stability, individual trajectories varied considerably. The finding that mild MIH cases universally showed OHRQoL decline while most severe cases improved challenges assumptions about treatment benefit distribution. This may reflect that children with mild MIH had less functional impairment initially, making any aesthetic concerns more prominent over time as they entered adolescence. The OHRQoL-DFA Relationship The inverse correlation between OHRQoL and DFA changes in 78% of participants represents a novel finding. This bidirectional relationship suggests successful treatment improving function may reduce anxiety, while negative experiences may simultaneously decrease OHRQoL and increase anxiety. This finding extends previous cross-sectional observations (McGrath and Bedi 2004; Luoto et al. 2009) by demonstrating the relationship longitudinally. Strengths of Mixed Methods Approach The qualitative analysis revealed important experiential dimensions not captured by standardised instruments. While quantitative measures showed minimal average change, qualitative data highlighted excellent clinical care but significant system-level challenges. This triangulation provides a more complete understanding of the MIH treatment journey. Limitations The 22% recruitment rate, while understandable given the 7-8 year follow-up period and pandemic context, limits generalisability. The remote data collection method, necessitated by COVID-19, prevented clinical examination. The MCDASf, validated for ages 8-12, was used beyond its intended age range to maintain measurement consistency. Clinical Implications Treatment planning should consider that functional improvements may be most beneficial for moderate/severe MIH Enhanced anxiety management strategies may be needed for extractions under local anaesthesia Service planning should consider the burden of travel time on families System improvements addressing waiting times and appointment availability could enhance patient experience despite excellent clinical care Treatment Modality Implications The association between permanent molar extractions under local anaesthesia and increased DFA warrants careful consideration. While numbers are small, this finding suggests the need for careful case selection and potentially enhanced anxiety management strategies for this procedure. Conversely, general anaesthesia showed the largest anxiety reductions in some cases, though with variable outcomes overall. Family Impact and Access The relationship between journey time and family impact highlights an important equity issue. With all families reporting zero financial burden within the UK's NHS system, travel distance emerged as the primary access barrier. This finding has implications for service planning and supports arguments for distributed specialist services (Wong and Kolokotsa 2004). Conclusions This long-term follow-up study demonstrates that MIH management outcomes are highly individual. The inverse relationship between OHRQoL and DFA changes, the potential anxiety impact of specific treatment modalities, and the importance of accessibility represent novel contributions to understanding MIH's long-term impact. The mixed-methods approach revealed that excellent clinical care can coexist with system-level challenges, emphasising the value of combining quantitative and qualitative methodologies in paediatric dental research. Future research should investigate factors predicting individual treatment response, optimal management strategies for different MIH severities, and interventions to support families facing access barriers. Prospective studies with higher retention rates and clinical follow-up examinations would strengthen the evidence base for long-term MIH management. Declarations Funding This research was conducted as part of a Doctorate in Paediatric Dentistry programme at the University of Leeds. Conflicts of Interest The authors declare no conflicts of interest. Ethics Approval NHS Research Ethics Committee Yorkshire and The Humber (REC: 21/PR/1069). Consent to Participate Written informed consent/assent was obtained from all participants and parents/guardians. Consent for Publication Participants consented to anonymous use of their data in publications. Availability of Data and Material Anonymised data are available from the corresponding author upon reasonable request. Author Contribution M.A.K.H. conceptualized and designed the study, obtained ethical approval, re-recruited participants, collected and managed all data, performed quantitative and qualitative analysis, and wrote the main manuscript text. R.B. main supervisor, supervised the study, provided methodological guidance, validated the analysis, and reviewed the thesis.S.F. supervised the study, provided methodological guidance, validated the analysis, and reviewed the thesis.K.K. supervised the qualitative aspect.B.D. provided supervisory oversight during early stages of the research.J.S. Supervised during early stages of the research. References Al-Bahar H (2017) The dental and orthodontic features, baseline anxiety and quality of life of children referred to a specialised centre for management of first permanent molars with Molar Incisor Hypomineralisation (MIH) or Caries. Thesis, University of Leeds Bekes K, Amend S, Priller J, Zamek C, Stamm T, Krämer N (2021) Changes in oral health-related quality of life after treatment of hypersensitive molar incisor hypomineralization-affected molars with a sealing. Clin Oral Investig 25:6449-6454 Broder HL, Wilson-Genderson M, Sischo L (2012) Reliability and validity testing for the Child Oral Health Impact Profile-Reduced (COHIP-SF 19). J Public Health Dent 72:302-312 Da Costa-Silva CM, Ambrosano GMB, Jeremias F, de Souza JF, Mialhe FL (2011) Increase in severity of molar-incisor hypomineralization and its relationship with the colour of enamel opacity: a prospective cohort study. Int J Paediatr Dent 21:333-341 Dantas-Neta NB, Moura LFAD, Cruz PF, Moura MS, Paiva SM, Martins CC, Lima MDM (2016) Impact of molar-incisor hypomineralization on oral health-related quality of life in schoolchildren. Braz Oral Res 30:e117 Dave M, Taylor G (2018) Global prevalence of molar incisor hypomineralisation. Evid Based Dent 19:78-79 Dias FMCS, Gradella CMF, Ferreira MC, Oliveira LB (2021) Molar-incisor hypomineralization: parent's and children's impact perceptions on the oral health-related quality of life. Eur Arch Paediatr Dent 22:273-282 Elhennawy K, Rajjoub O, Reissmann DR, Doueiri MS, Hamad R, Sierwald I, Wiedemann V, Bekes K, Jost-Brinkmann PG (2022) The association between molar incisor hypomineralization and oral health-related quality of life: a cross-sectional study. Clin Oral Investig 26:4071-4077 Fragelli CM, Souza JF, Jeremias F, Cordeiro RCL, Santos-Pinto L (2015) Molar incisor hypomineralization (MIH): conservative treatment management to restore affected teeth. Braz Oral Res 29:1-7 Howard KE, Freeman R (2007) Reliability and validity of a faces version of the Modified Child Dental Anxiety Scale. Int J Paediatr Dent 17:281-288 Jälevik B, Klingberg G (2012) Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls - a longitudinal study. Int J Paediatr Dent 22:85-91 Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N (2016) Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent 17:123-129 Krippendorff K (2018) Content analysis: an introduction to its methodology, 4th edn. SAGE, Los Angeles Linner T, Khazaei Y, Bücher K, Pfisterer J, Hickel R, Kühnisch J (2021) Hypersensitivity in teeth affected by molar-incisor hypomineralization (MIH). Sci Rep 11:17922 Locker D, Jokovic A, Stephens M, Kenny D, Tompson B, Guyatt G (2002) Family impact of child oral and oro-facial conditions. Community Dent Oral Epidemiol 30:438-448 Lopes LB, Machado V, Mascarenhas P, Mendes JJ, Botelho J (2021) The prevalence of molar-incisor hypomineralization: a systematic review and meta-analysis. Sci Rep 11:22405 Luoto A, Lahti S, Nevanperä T, Tolvanen M, Locker D (2009) Oral-health-related quality of life among children with and without dental fear. Int J Paediatr Dent 19:115-120 Lygidakis NA, Garot E, Somani C, Taylor GD, Rouas P, Wong FSL (2022) Best clinical practice guidance for clinicians dealing with children presenting with molar-incisor-hypomineralisation (MIH): an updated European Academy of Paediatric Dentistry policy document. Eur Arch Paediatr Dent 23:3-21 McGrath C, Bedi R (2004) The association between dental anxiety and oral health-related quality of life in Britain. Community Dent Oral Epidemiol 32:67-72 Rodd HD, Boissonade FM, Day PF (2007) Pulpal status of hypomineralized permanent molars. Pediatr Dent 29:514-520 Schwendicke F, Elhennawy K, Reda S, Bekes K, Manton DJ, Krois J (2018) Global burden of molar incisor hypomineralization. J Dent 68:10-18 Wong FSL, Kolokotsa K (2004) The cost of treating children and adolescents with injuries to their permanent incisors at a dental hospital in the United Kingdom. Dent Traumatol 20:327-333 Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-8200677","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":559013526,"identity":"fc238e84-a36e-4435-af23-d0c7dd67ae75","order_by":0,"name":"Mohammad A Kh Hussein","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAAx0lEQVRIiWNgGAWjYFACHgbGxoYaO34GBjaStBxLlmwgUQsz44YDxGrRbe89+HHmDjZm4xvJzx58qGCQ5xc7gF+L2ZlzyZIbz8jwmd1IMzeccYbBcObsBAJabuQYSD5sY2M2u5FgJs3bxpBgcJuwFuOfD9uYGTfPSP9GtBYzyY1ALRskcoi15cwZM8uZbceSJc68KZOccUaCCL8c7zG+2dsGjMr29G0SHyps5PmlCWhBAAGwSglilYMA/wFSVI+CUTAKRsFIAgCaPkWAOTse0gAAAABJRU5ErkJggg==","orcid":"","institution":"","correspondingAuthor":true,"prefix":"","firstName":"Mohammad","middleName":"A Kh","lastName":"Hussein","suffix":""}],"badges":[],"createdAt":"2025-11-25 08:23:16","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-8200677/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-8200677/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":98378020,"identity":"9edca0d3-c2b5-4554-805c-bf950b8edca7","added_by":"auto","created_at":"2025-12-17 07:20:37","extension":"docx","order_by":0,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":34979,"visible":true,"origin":"","legend":"","description":"","filename":"ManusriptNovember2025.docx","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/9b6ffd27c691d16f41f5bb94.docx"},{"id":98378021,"identity":"e1605a16-0e57-4ecd-a277-a98cc8732708","added_by":"auto","created_at":"2025-12-17 07:20:37","extension":"json","order_by":1,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":4942,"visible":true,"origin":"","legend":"","description":"","filename":"03085f13c3664f31a5835b35d72e583a.json","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/3c12dc90fccff33fe5cdc279.json"},{"id":98439802,"identity":"28e27fc4-64eb-4839-8dec-70b8ddcd6077","added_by":"auto","created_at":"2025-12-17 17:02:57","extension":"xml","order_by":2,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":43185,"visible":true,"origin":"","legend":"","description":"","filename":"03085f13c3664f31a5835b35d72e583a1enriched.xml","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/6444bbc09c11153baa036600.xml"},{"id":98439737,"identity":"19e82fd8-9ae3-49ae-8542-56e3c0ec19bc","added_by":"auto","created_at":"2025-12-17 17:02:51","extension":"xml","order_by":7,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":40129,"visible":true,"origin":"","legend":"","description":"","filename":"03085f13c3664f31a5835b35d72e583a1structuring.xml","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/95b0559e1eba82ac921b376a.xml"},{"id":98378019,"identity":"a0e5c797-d8a4-47c6-a69d-493f7998e2e0","added_by":"auto","created_at":"2025-12-17 07:20:37","extension":"html","order_by":8,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":49155,"visible":true,"origin":"","legend":"","description":"","filename":"earlyproof.html","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/4b97801f6600410e010a003e.html"},{"id":98378016,"identity":"275186b6-7042-4b93-8d15-23debd18b9ef","added_by":"auto","created_at":"2025-12-17 07:20:37","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":55046,"visible":true,"origin":"","legend":"\u003cp\u003eDifference in FIS total scores and the time of the journey to LDI.\u003c/p\u003e","description":"","filename":"1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/a944ea02716a7ba4f763b9ca.jpg"},{"id":98623009,"identity":"f252d8c9-63e3-4a68-8f74-870818213b78","added_by":"auto","created_at":"2025-12-19 17:04:01","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":771514,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-8200677/v1/0983cf12-798b-4919-8a1d-e55a110bc6f2.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Long-term impact of molar-incisor hypomineralisation and its management on children and their families: an 8-year follow-up study with mixed methods analysis","fulltext":[{"header":"Introduction","content":"\u003cp\u003eMolar-incisor hypomineralisation (MIH) affects approximately 13\u0026ndash;14% of children globally (Dave and Taylor 2018; Schwendicke et al. 2018; Lopes et al. 2021), presenting significant clinical challenges including hypersensitivity, post-eruptive breakdown, and aesthetic concerns (Linner et al. 2021; Rodd et al. 2007). While immediate treatment outcomes have been documented (Fragelli et al. 2015; Bekes et al. 2021), long-term impacts on affected children and their families remain poorly understood. The European Academy of Paediatric Dentistry's 2022 clinical guidelines specifically identified the need for longitudinal research examining psychosocial and economic aspects of MIH (Lygidakis et al. 2022).\u003c/p\u003e \u003cp\u003ePrevious cross-sectional studies have reported conflicting findings regarding MIH's impact on oral health-related quality of life (OHRQoL), with some demonstrating significant negative effects (Dantas-Neta et al. 2016; Elhennawy et al. 2022) while others found minimal impact (Dias et al. 2021). Similarly, evidence regarding dental fear and anxiety (DFA) in MIH-affected children remains inconsistent (J\u0026auml;levik and Klingberg 2012; Kosma et al. 2016). Critically, no previous studies have prospectively tracked the same cohort from pre-treatment through long-term post-treatment follow-up using validated instruments, nor have they incorporated qualitative analysis of patient and family experiences.\u003c/p\u003e \u003cp\u003eThis study addresses these gaps by presenting 7\u0026ndash;8 year follow-up data from a prospectively recruited cohort, combining quantitative assessment of OHRQoL and DFA changes with qualitative exploration of treatment experiences. We aimed to: (1) evaluate long-term changes in OHRQoL and DFA following specialist MIH management; (2) assess the impact on families; (3) identify factors associated with outcomes; and (4) explore patient/parent perspectives through qualitative analysis.\u003c/p\u003e"},{"header":"Materials and Methods","content":"\u003cp\u003e\u003cstrong\u003eStudy Design and Ethical Approval\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis prospective follow-up study investigated a cohort originally recruited during 2015-2017 at Leeds Dental Institute, UK. Ethical approval was obtained from NHS Research Ethics Committee Yorkshire and The Humber (REC: 21/PR/1069) and Leeds Teaching Hospitals NHS Trust (R\u0026amp;I: DT22/148475).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eParticipants\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eFrom 82 children with MIH in the original cohort (Al-Bahar 2017), all were invited to participate via postal invitation followed by telephone contact. The COVID-19 pandemic necessitated remote data collection methods. Inclusion criteria were: previous participation in the baseline study, confirmed MIH diagnosis, and consent/assent from both participant and parent/guardian.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eData Collection\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQuantitative Measures\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eParticipants completed the same validated instruments used at baseline:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003e\u003cstrong\u003eCOHIP-SF19\u003c/strong\u003e (Broder et al. 2012): 19-item scale assessing OHRQoL across three domains (oral health, functional well-being, socio-emotional well-being). Scores range 0-76, with higher scores indicating better OHRQoL.\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eMCDASf\u003c/strong\u003e (Howard and Freeman 2007): 8-item scale measuring dental anxiety using facial expressions. Scores range 8-40, with higher scores indicating greater anxiety.\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eParents completed:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003e\u003cstrong\u003eFamily Impact Scale (FIS)\u003c/strong\u003e (Locker et al. 2002): 14-item scale assessing impact across four domains (parental activities, parental emotions, family conflict, financial burden). Scores range 0-56, with higher scores indicating greater impact.\u003c/li\u003e\n \u003cli\u003eTransportation questionnaire: mode, duration, cost, and perceived difficulty of journeys to the specialist centre.\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003e\u003cstrong\u003eClinical Data\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eElectronic and physical paper patient records were systematically reviewed to extract: MIH severity at baseline (mild/moderate/severe per Da Costa-Silva et al. 2011 criteria), treatment modalities (local anaesthesia/inhalation sedation/general anaesthesia), procedures performed, and number of visits.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQualitative Data\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eOpen-ended questions explored experiences of care:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003eParticipants: difficulties encountered, positive aspects, suggestions for improvement\u003c/li\u003e\n \u003cli\u003eParents: what was done well, areas for improvement, additional comments\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003e\u003cstrong\u003eData Analysis\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQuantitative Analysis\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eDescriptive statistics were calculated for all measures. Individual change scores were computed (follow-up minus baseline). Relationships between variables were explored graphically due to small sample size. Data were analysed using Microsoft Excel.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQualitative Analysis\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eContent analysis followed established methodology (Krippendorff 2018). Two researchers independently reviewed responses, identifying recurring themes and developing codes. NVivo 1.7.1 facilitated systematic coding. Themes were refined through discussion until consensus was reached.\u003c/p\u003e"},{"header":"Results","content":"\u003cp\u003e\u003cstrong\u003eParticipant Characteristics\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eEighteen participants were recruited (22% response rate): 13 males, 5 females; mean age 15.6 years (range 14-17) at follow-up; mean 7.5 years since baseline. At baseline: 11 had severe MIH, 4 moderate, 3 mild. Treatment modalities included: local anaesthesia only (n=5), general anaesthesia only (n=6), combined approaches (n=3), no active intervention (n=4). Eleven participants underwent extractions, 7 received restorations.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQuantitative Outcomes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOHRQoL Changes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eOverall COHIP-SF19 scores showed minimal mean change (baseline: 56.3\u0026plusmn;9.5, follow-up: 53.6\u0026plusmn;12.3, mean change: -2.6). However, individual changes ranged from +18 to -23 points. Domain analysis revealed:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003e\u003cstrong\u003eFunctional well-being\u003c/strong\u003e: 12/18 improved (mean change: +1.0)\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eOral health\u003c/strong\u003e: 7/18 improved (mean change: -0.4)\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eSocio-emotional well-being\u003c/strong\u003e: 8/18 improved (mean change: -3.1)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eNotably, all three mild MIH cases showed OHRQoL decline, while 7/11 severe cases improved.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDFA Changes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eMCDASf scores remained stable overall (baseline: 20.1\u0026plusmn;6.8, follow-up: 20.5\u0026plusmn;7.4, mean change: +0.5), but individual changes ranged from -12 to +24. The two largest anxiety increases occurred in participants receiving permanent molar extractions under local anaesthesia (+24 and +10 points).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eRelationship Between OHRQoL and DFA\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eChanges in COHIP-SF19 and MCDASf were inversely correlated in 14/18 participants: improved OHRQoL accompanied reduced anxiety or vice versa.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eTable1\u0026nbsp;\u003c/strong\u003eDifferences in COHIP-SF19 total scores and in MCDASf total scores, and their changes between baseline and follow-up studies, ranked according to the difference in total COHIP-SF19.\u003c/p\u003e\n\u003ctable border=\"1\" cellspacing=\"0\" cellpadding=\"0\"\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eParticipant ID\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eDifference in COHIP-SF19 total score\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eDifference in MCDASf total score\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e108\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e18\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-10\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e28\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e6\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-4\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e46\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e6\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e24\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e65\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e6\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e43\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-2\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e66\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e63\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-8\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e12\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e97\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-12\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e30\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-2\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e51\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-5\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e107\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e10\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e49\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e99\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e100\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-19\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e8\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-23\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e60\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e-23\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 181px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003e\u003cu\u003eNote\u003c/u\u003e\u003c/p\u003e\n\u003cp\u003e- A positive change (increase) in total COHIP SF-19 is indicative of an improvement in OH QoL (indicated in green). A decline in COHIP-SF19 score is indicative of a decrease in OH QoL (Red).\u003c/p\u003e\n\u003cp\u003e- A positive change (increase in MCDASf score indicates an increase in dental anxiety (indicated in Red). \u0026nbsp; A decrease in MCDASf score is indicative of a decrease in dental anxiety (Green).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFamily Impact\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eMean FIS score was 6.0\u0026plusmn;6.7 (range 0-25), indicating generally low impact. All families reported zero financial burden. Impact increased with journey time: families travelling \u0026gt;60 minutes had mean FIS 13.7 versus 3.8 for \u0026lt;60 minutes. Parental activities (mean 2.8) and emotions (mean 2.0) were most affected domains.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eQualitative Findings\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eTwo main themes emerged from content analysis:\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eTheme 1: Communication Excellence\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eParents and participants consistently praised communication:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;Everything was explained in detail to myself and my child\u0026quot;\u003c/em\u003e (Parent-028)\u003c/li\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;Great explanation of what was going on. Made [child] feel really comfortable\u0026quot;\u003c/em\u003e (Parent-063)\u003c/li\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;Staff were friendly\u0026quot;\u003c/em\u003e (Multiple respondents)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003e\u003cstrong\u003eTheme 2: System Challenges\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWaiting times and appointment availability were primary concerns:\u003c/p\u003e\n\u003cul type=\"disc\"\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;Waited long time for appointments. Follow-up slow. Had to ring to chase appointments\u0026quot;\u003c/em\u003e (Parent-030)\u003c/li\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;The availability of appointments is quite limiting. I feel like treatment has taken longer due to this\u0026quot;\u003c/em\u003e (Parent-063)\u003c/li\u003e\n \u003cli\u003e\u003cem\u003e\u0026quot;Reduce waiting time as this increases anxiety\u0026quot;\u003c/em\u003e (Parent-108)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eDespite challenges, 17/18 families reported satisfaction with overall care. While one family found the experience to be neutral.\u0026nbsp;\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThis study presents the first long-term prospective follow-up of MIH-affected children using validated pre- and post-treatment measures combined with qualitative analysis, addressing a critical gap identified by EAPD guidelines (Lygidakis et al. 2022).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eIndividual Variability in Outcomes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWhile group means suggested stability, individual trajectories varied considerably. The finding that mild MIH cases universally showed OHRQoL decline while most severe cases improved challenges assumptions about treatment benefit distribution. This may reflect that children with mild MIH had less functional impairment initially, making any aesthetic concerns more prominent over time as they entered adolescence.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eThe OHRQoL-DFA Relationship\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe inverse correlation between OHRQoL and DFA changes in 78% of participants represents a novel finding. This bidirectional relationship suggests successful treatment improving function may reduce anxiety, while negative experiences may simultaneously decrease OHRQoL and increase anxiety. This finding extends previous cross-sectional observations (McGrath and Bedi 2004; Luoto et al. 2009) by demonstrating the relationship longitudinally.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eStrengths of Mixed Methods Approach\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe qualitative analysis revealed important experiential dimensions not captured by standardised instruments. While quantitative measures showed minimal average change, qualitative data highlighted excellent clinical care but significant system-level challenges. This triangulation provides a more complete understanding of the MIH treatment journey.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eLimitations\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe 22% recruitment rate, while understandable given the 7-8 year follow-up period and pandemic context, limits generalisability. The remote data collection method, necessitated by COVID-19, prevented clinical examination. The MCDASf, validated for ages 8-12, was used beyond its intended age range to maintain measurement consistency.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eClinical Implications\u003c/strong\u003e\u003c/p\u003e\n\u003col start=\"1\" type=\"1\"\u003e\n \u003cli\u003eTreatment planning should consider that functional improvements may be most beneficial for moderate/severe MIH\u003c/li\u003e\n \u003cli\u003eEnhanced anxiety management strategies may be needed for extractions under local anaesthesia\u003c/li\u003e\n \u003cli\u003eService planning should consider the burden of travel time on families\u003c/li\u003e\n \u003cli\u003eSystem improvements addressing waiting times and appointment availability could enhance patient experience despite excellent clinical care\u003c/li\u003e\n\u003c/ol\u003e\n\u003cp\u003e\u003cstrong\u003eTreatment Modality Implications\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe association between permanent molar extractions under local anaesthesia and increased DFA warrants careful consideration. While numbers are small, this finding suggests the need for careful case selection and potentially enhanced anxiety management strategies for this procedure. Conversely, general anaesthesia showed the largest anxiety reductions in some cases, though with variable outcomes overall.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFamily Impact and Access\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe relationship between journey time and family impact highlights an important equity issue. With all families reporting zero financial burden within the UK's NHS system, travel distance emerged as the primary access barrier. This finding has implications for service planning and supports arguments for distributed specialist services (Wong and Kolokotsa 2004).\u003c/p\u003e"},{"header":"Conclusions","content":"\u003cp\u003eThis long-term follow-up study demonstrates that MIH management outcomes are highly individual. The inverse relationship between OHRQoL and DFA changes, the potential anxiety impact of specific treatment modalities, and the importance of accessibility represent novel contributions to understanding MIH's long-term impact. The mixed-methods approach revealed that excellent clinical care can coexist with system-level challenges, emphasising the value of combining quantitative and qualitative methodologies in paediatric dental research.\u003c/p\u003e \u003cp\u003eFuture research should investigate factors predicting individual treatment response, optimal management strategies for different MIH severities, and interventions to support families facing access barriers. Prospective studies with higher retention rates and clinical follow-up examinations would strengthen the evidence base for long-term MIH management.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e This research was conducted as part of a Doctorate in Paediatric Dentistry programme at the University of Leeds.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConflicts of Interest\u003c/strong\u003e The authors declare no conflicts of interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics Approval\u003c/strong\u003e NHS Research Ethics Committee Yorkshire and The Humber (REC: 21/PR/1069).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent to Participate\u003c/strong\u003e Written informed consent/assent was obtained from all participants and parents/guardians.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for Publication\u003c/strong\u003e Participants consented to anonymous use of their data in publications.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of Data and Material\u003c/strong\u003e Anonymised data are available from the corresponding author upon reasonable request.\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eM.A.K.H. conceptualized and designed the study, obtained ethical approval, re-recruited participants, collected and managed all data, performed quantitative and qualitative analysis, and wrote the main manuscript text. R.B. main supervisor, supervised the study, provided methodological guidance, validated the analysis, and reviewed the thesis.S.F. supervised the study, provided methodological guidance, validated the analysis, and reviewed the thesis.K.K. supervised the qualitative aspect.B.D. provided supervisory oversight during early stages of the research.J.S. Supervised during early stages of the research.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eAl-Bahar H (2017) The dental and orthodontic features, baseline anxiety and quality of life of children referred to a specialised centre for management of first permanent molars with Molar Incisor Hypomineralisation (MIH) or Caries. Thesis, University of Leeds\u003c/li\u003e\n\u003cli\u003eBekes K, Amend S, Priller J, Zamek C, Stamm T, Kr\u0026auml;mer N (2021) Changes in oral health-related quality of life after treatment of hypersensitive molar incisor hypomineralization-affected molars with a sealing. Clin Oral Investig 25:6449-6454\u003c/li\u003e\n\u003cli\u003eBroder HL, Wilson-Genderson M, Sischo L (2012) Reliability and validity testing for the Child Oral Health Impact Profile-Reduced (COHIP-SF 19). J Public Health Dent 72:302-312\u003c/li\u003e\n\u003cli\u003eDa Costa-Silva CM, Ambrosano GMB, Jeremias F, de Souza JF, Mialhe FL (2011) Increase in severity of molar-incisor hypomineralization and its relationship with the colour of enamel opacity: a prospective cohort study. Int J Paediatr Dent 21:333-341\u003c/li\u003e\n\u003cli\u003eDantas-Neta NB, Moura LFAD, Cruz PF, Moura MS, Paiva SM, Martins CC, Lima MDM (2016) Impact of molar-incisor hypomineralization on oral health-related quality of life in schoolchildren. Braz Oral Res 30:e117\u003c/li\u003e\n\u003cli\u003eDave M, Taylor G (2018) Global prevalence of molar incisor hypomineralisation. Evid Based Dent 19:78-79\u003c/li\u003e\n\u003cli\u003eDias FMCS, Gradella CMF, Ferreira MC, Oliveira LB (2021) Molar-incisor hypomineralization: parent\u0026apos;s and children\u0026apos;s impact perceptions on the oral health-related quality of life. Eur Arch Paediatr Dent 22:273-282\u003c/li\u003e\n\u003cli\u003eElhennawy K, Rajjoub O, Reissmann DR, Doueiri MS, Hamad R, Sierwald I, Wiedemann V, Bekes K, Jost-Brinkmann PG (2022) The association between molar incisor hypomineralization and oral health-related quality of life: a cross-sectional study. Clin Oral Investig 26:4071-4077\u003c/li\u003e\n\u003cli\u003eFragelli CM, Souza JF, Jeremias F, Cordeiro RCL, Santos-Pinto L (2015) Molar incisor hypomineralization (MIH): conservative treatment management to restore affected teeth. Braz Oral Res 29:1-7\u003c/li\u003e\n\u003cli\u003eHoward KE, Freeman R (2007) Reliability and validity of a faces version of the Modified Child Dental Anxiety Scale. Int J Paediatr Dent 17:281-288\u003c/li\u003e\n\u003cli\u003eJ\u0026auml;levik B, Klingberg G (2012) Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls - a longitudinal study. Int J Paediatr Dent 22:85-91\u003c/li\u003e\n\u003cli\u003eKosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N (2016) Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent 17:123-129\u003c/li\u003e\n\u003cli\u003eKrippendorff K (2018) Content analysis: an introduction to its methodology, 4th edn. SAGE, Los Angeles\u003c/li\u003e\n\u003cli\u003eLinner T, Khazaei Y, B\u0026uuml;cher K, Pfisterer J, Hickel R, K\u0026uuml;hnisch J (2021) Hypersensitivity in teeth affected by molar-incisor hypomineralization (MIH). Sci Rep 11:17922\u003c/li\u003e\n\u003cli\u003eLocker D, Jokovic A, Stephens M, Kenny D, Tompson B, Guyatt G (2002) Family impact of child oral and oro-facial conditions. Community Dent Oral Epidemiol 30:438-448\u003c/li\u003e\n\u003cli\u003eLopes LB, Machado V, Mascarenhas P, Mendes JJ, Botelho J (2021) The prevalence of molar-incisor hypomineralization: a systematic review and meta-analysis. Sci Rep 11:22405\u003c/li\u003e\n\u003cli\u003eLuoto A, Lahti S, Nevanper\u0026auml; T, Tolvanen M, Locker D (2009) Oral-health-related quality of life among children with and without dental fear. Int J Paediatr Dent 19:115-120\u003c/li\u003e\n\u003cli\u003eLygidakis NA, Garot E, Somani C, Taylor GD, Rouas P, Wong FSL (2022) Best clinical practice guidance for clinicians dealing with children presenting with molar-incisor-hypomineralisation (MIH): an updated European Academy of Paediatric Dentistry policy document. Eur Arch Paediatr Dent 23:3-21\u003c/li\u003e\n\u003cli\u003eMcGrath C, Bedi R (2004) The association between dental anxiety and oral health-related quality of life in Britain. Community Dent Oral Epidemiol 32:67-72\u003c/li\u003e\n\u003cli\u003eRodd HD, Boissonade FM, Day PF (2007) Pulpal status of hypomineralized permanent molars. Pediatr Dent 29:514-520\u003c/li\u003e\n\u003cli\u003eSchwendicke F, Elhennawy K, Reda S, Bekes K, Manton DJ, Krois J (2018) Global burden of molar incisor hypomineralization. J Dent 68:10-18\u003c/li\u003e\n\u003cli\u003eWong FSL, Kolokotsa K (2004) The cost of treating children and adolescents with injuries to their permanent incisors at a dental hospital in the United Kingdom. Dent Traumatol 20:327-333\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"MIH, OHRQoL, DFA, FIS, Long-term follow-up, Mixed methods","lastPublishedDoi":"10.21203/rs.3.rs-8200677/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-8200677/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003ePurpose\u003c/h2\u003e \u003cp\u003eTo investigate long-term changes in oral health-related quality of life (OHRQoL) and dental fear/anxiety (DFA) following specialist management of molar-incisor hypomineralisation (MIH), assess family impact, and explore patient/parent experiences through qualitative analysis.\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e \u003cp\u003eThis prospective follow-up study re-recruited participants from a 2015\u0026ndash;2017 baseline cohort of children with MIH referred to a UK specialist centre. Participants completed the same validated instruments used at baseline: Child Oral Health Impact Profile-Short Form 19 (COHIP-SF19) and Modified Child Dental Anxiety Scale-faces version (MCDASf). Additionally, parents completed the Family Impact Scale (FIS). Clinical records were reviewed retrospectively. Qualitative content analysis was performed on open-ended responses about treatment experiences. Changes were analysed using descriptive statistics and thematic analysis.\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e \u003cp\u003eEighteen participants (22% re-recruitment rate; mean age 15.6 years; 13 males) were followed up 7\u0026ndash;8 years post-baseline. At baseline, 11 had severe, 4 moderate, and 3 mild MIH. Treatment modalities included local anaesthesia (n\u0026thinsp;=\u0026thinsp;9), general anaesthesia (n\u0026thinsp;=\u0026thinsp;8), and inhalation sedation (n\u0026thinsp;=\u0026thinsp;2). Mean COHIP-SF19 scores showed minimal overall change (baseline: 56.3, follow-up: 53.6), with functional well-being showing greatest improvement. MCDASf scores remained stable (baseline: 20.1, follow-up: 20.5). However, individual variation was substantial, with OHRQoL and DFA changes inversely correlated in 14/18 participants. Permanent molar extractions under local anaesthesia were associated with the largest DFA increases. Family impact was generally low (mean FIS: 6/56) but increased with journey time to the specialist centre (\u0026gt;\u0026thinsp;60 minutes). Qualitative analysis revealed two main themes: positive communication experiences and system challenges (waiting times, appointment availability).\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e \u003cp\u003eThis long-term follow-up study demonstrates heterogeneous outcomes following MIH management. While group means suggest stability, individual trajectories varied considerably. The inverse relationship between OHRQoL and DFA changes, the potential anxiety impact of extractions under local anaesthesia, and the burden of travel distance represent novel findings requiring further investigation. The mixed-methods approach provided valuable insights into patient/family experiences that quantitative measures alone would miss.\u003c/p\u003e","manuscriptTitle":"Long-term impact of molar-incisor hypomineralisation and its management on children and their families: an 8-year follow-up study with mixed methods analysis","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-12-17 07:20:33","doi":"10.21203/rs.3.rs-8200677/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"d29a9d67-e373-4713-8a2d-98c76cfbf834","owner":[],"postedDate":"December 17th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2025-12-17T14:24:51+00:00","versionOfRecord":[],"versionCreatedAt":"2025-12-17 07:20:33","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-8200677","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-8200677","identity":"rs-8200677","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}