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Do patients with molar incisor hypomineralization have more dental anxiety and behavior management problems? A systematic review with meta-analysis

Abstract

This systematic review evaluated the available evidence on whether children with molar incisor hypomineralization (MIH) have more dental fear and anxiety (DFA) and dental behavior management problems (DBMPs) than those without MIH (Prospero CDR42020203851). Unrestricted searches were performed in PubMed, Scopus, Web of Science, Lilacs, BBO, Embase, Cochrane Library, APA PsycINFO, Open Grey, and Google Scholar. Observational studies evaluating DFA and/or DBMPs in patients with and without MIH were eligible. Reviews, case reports, interventional studies, and those based on questionnaires to dentists were excluded. The methodological quality assessment was based on the Newcastle-Ottawa Scale. Random-effects meta-analyses were conducted to synthesize data on DFA. The certainty of evidence was performed according to GRADE. Seven studies that evaluated a total of 3,805 patients were included. All of them presented methodological issues, mainly in the comparability domain. Most studies observed no significant difference in DFA between children with and without MIH. The meta-analysis did not show a significant effect of MIH on the standardized units for the DFA scores (SMD = 0.03; 95%CI: -0.06–0.12; p = 0.53; I2 = 0%). Synthesis including only the results for severe cases of MIH also did not show a significant effect of the condition on DFA scores (MD = 8.68; 95%CI: -8.64–26.00; p = 0.33; I2 = 93%). Two articles found DBMPs were significantly more frequent in patients with MIH. The overall certainty of evidence was very low for both outcomes assessed. The current evidence suggests no difference in DFA between children with and without MIH; DBMPs are more common in patients with MIH. This information should be viewed with caution because of the very low quality evidence obtained.

Dental Enamel Hypoplasia; Dental Anxiety; Behavior

Introduction

The terms dental fear and dental anxiety represent different progressive degrees of the same psychological condition and have been used indistinctly in scientific literature.11. Klinberg G. Dental anxiety and behaviour management problems in paediatric dentistry: a review of background factors and diagnostics. Eur Arch Paediatr Dent. 2008 Feb;9(1 Suppl 1):11-5. https://doi.org/10.1007/BF03262650
https://doi.org/10.1007/BF03262650...
The term dental fear and anxiety (DFA) is used to describe strong negative emotions associated with dental treatment among children and adolescents.22. Klingberg G, Broberg AG. Dental fear/anxiety and dental behaviour management problems in children and adolescents: a review of prevalence and concomitant psychological factors. Int J Paediatr Dent. 2007 Nov;17(6):391-406. https://doi.org/10.1111/j.1365-263X.2007.00872.x
https://doi.org/10.1111/j.1365-263X.2007...
Dental behavior management problems (DBMPs), defined as uncooperative and disruptive behaviors resulting in delay of treatment or making it impossible,33. Klingberg G, Vannas Löfqvist L, Bjarnason S, Norén JG. Dental behavior management problems in Swedish children. Community Dent Oral Epidemiol. 1994 Jun;22(3):201-5. https://doi.org/10.1111/j.1600-0528.1994.tb01841.x
https://doi.org/10.1111/j.1600-0528.1994...
are more frequently observed in patients with DFA.44. Gao S, Lu J, Li P, Yu D, Zhao W. Prevalence and risk factors of children’s dental anxiety in China: a longitudinal study. BMJ Open. 2021 Apr;11(4):e043647. https://doi.org/10.1136/bmjopen-2020-043647
https://doi.org/10.1136/bmjopen-2020-043...
, 55. Pai R, Mandroli P, Benni D, Pujar P. Prospective analysis of factors associated with dental behavior management problems, in children aged 7-11 years. J Indian Soc Pedod Prev Dent. 2015;33(4):312-8. https://doi.org/10.4103/0970-4388.165684
https://doi.org/10.4103/0970-4388.165684...

DFA is a problem frequently faced by pediatric dentists, interfering with the management and dental treatment of children.66. Cohen SM, Fiske J, Newton JT. The impact of dental anxiety on daily living. Br Dent J. 2000 Oct;189(7):385-90. https://doi.org/10.1038/sj.bdj.4800777
https://doi.org/10.1038/sj.bdj.4800777...
, 77. Armfield JM. What goes around comes around: revisiting the hypothesized vicious cycle of dental fear and avoidance. Community Dent Oral Epidemiol. 2013 Jun;41(3):279-87. https://doi.org/10.1111/cdoe.12005
https://doi.org/10.1111/cdoe.12005...
Anxious patients usually delay dental treatment and routinely miss the appointments, which can lead to worsening of oral health and the need for more complex procedures.66. Cohen SM, Fiske J, Newton JT. The impact of dental anxiety on daily living. Br Dent J. 2000 Oct;189(7):385-90. https://doi.org/10.1038/sj.bdj.4800777
https://doi.org/10.1038/sj.bdj.4800777...

The reason why patients develop DFA is still unclear. Some factors may be related to its presence, such as previous experience with dental caries and dental pain.88. Torriani DD, Ferro RL, Bonow ML, Santos IS, Matijasevich A, Barros AJ, et al. Dental caries is associated with dental fear in childhood: findings from a birth cohort study. Caries Res. 2014;48(4):263-70. https://doi.org/10.1159/000356306
https://doi.org/10.1159/000356306...

9. Viswanath D, Krishna AV. Correlation between dental anxiety, Sense of Coherence (SOC) and dental caries in school children from Bangalore North: a cross-sectional study. J Indian Soc Pedod Prev Dent. 2015;33(1):15-8. https://doi.org/10.4103/0970-4388.148962
https://doi.org/10.4103/0970-4388.148962...

10. Silveira ER, Goettems ML, Demarco FF, Azevedo MS. Clinical and individual variables in children’s dental fear: A school - Based investigation. Braz Dent J. 2017;28(3):398-404. https://doi.org/10.1590/0103-6440201601265
https://doi.org/10.1590/0103-64402016012...
- 1111. Abanto J, Vidigal EA, Carvalho TS, Sá SN, Bönecker M. Factors for determining dental anxiety in preschool children with severe dental caries. Braz Oral Res. 2017 Jan;31(0):e13. https://doi.org/10.1590/1807-3107bor-2017.vol31.0013
https://doi.org/10.1590/1807-3107bor-201...
However, a recent study has not found any association between dental caries and dental fear,1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
while another study has found that dental anxiety was influenced by previous experiences with dental caries in the primary dentition.1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
The presence of MIH did not seem to increase DFA, but it had a negative impact on the quality of life of children and adolescents.1414. Jälevik B, Sabel N, Robertson A. Can molar incisor hypomineralization cause dental fear and anxiety or influence the oral health-related quality of life in children and adolescents?-a systematic review. Eur Arch Paediatr Dent. 2022 Feb;23(1):65-78. https://doi.org/10.1007/s40368-021-00631-4
https://doi.org/10.1007/s40368-021-00631...

DBPMs have posed a constant challenge to pediatric dentists. Failure to handle a child with disruptive behavior may compromise dental treatment. To avoid this problem, it is useful to identify children at risk for DBMPs in order to develop an appropriate management strategy. Previous unpleasant dental or medical experience and awareness of existing dental problems are factors that usually cause a negative impact on children’s behavior. Conversely, an appropriate approach to these children, shorter appointments, and less complex procedures favor a better behavior in the dental chair.44. Gao S, Lu J, Li P, Yu D, Zhao W. Prevalence and risk factors of children’s dental anxiety in China: a longitudinal study. BMJ Open. 2021 Apr;11(4):e043647. https://doi.org/10.1136/bmjopen-2020-043647
https://doi.org/10.1136/bmjopen-2020-043...
, 55. Pai R, Mandroli P, Benni D, Pujar P. Prospective analysis of factors associated with dental behavior management problems, in children aged 7-11 years. J Indian Soc Pedod Prev Dent. 2015;33(4):312-8. https://doi.org/10.4103/0970-4388.165684
https://doi.org/10.4103/0970-4388.165684...

Molar incisor hypomineralization (MIH) is a qualitative defect that causes the affected enamel to be more porous and fragile, tending to fracture under masticatory forces and to expose the underlying dentin.1515. Beentjes VE, Weerheijm KL, Groen HJ. Factors involved in the aetiology of molar-incisor hypomineralisation (MIH). Eur J Paediatr Dent. 2002 Mar;3(1):9-13. PMID:12871011 Children with MIH usually complain about tooth hypersensitivity, tend to avoid toothbrushing, which predisposes to bacterial plaque accumulation and contributes to the development of dental caries.1616. Lygidakis NA, Wong F, Jälevik B, Vierrou AM, Alaluusua S, Espelid I. Best clinical practice guidance for clinicians dealing with children presenting with Molar-Incisor-Hypomineralisation (MIH): an EAPD Policy Document. Eur Arch Paediatr Dent. 2021;22(5)851-860. https://doi.org10.1007/s40368-021-00649-8
https://doi.org10.1007/s40368-021-00649-...

Patients with MIH are usually reluctant to open their mouths and react intensively to air jet even when it is used only for dental examination.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. The treatment can be painful as it is more difficult to obtain an effective anesthesia due to a chronic inflammation of the pulp cells.1818. Rodd HD, Morgan CR, Day PF, Boissonade FM. Pulpal expression of TRPV1 in molar incisor hypomineralisation. Eur Arch Paediatr Dent. 2007 Dec;8(4):184-8. https://doi.org/10.1007/BF03262594
https://doi.org/10.1007/BF03262594...
Furthermore, the altered prismatic morphology of hypomineralized enamel impairs bonding and can lead to loss of fillings.1919. Krämer N, Bui Khac NN, Lücker S, Stachniss V, Frankenberger R. Bonding strategies for MIH-affected enamel and dentin. Dent Mater. 2018 Feb;34(2):331-40. https://doi.org/10.1016/j.dental.2017.11.015
https://doi.org/10.1016/j.dental.2017.11...
Molars severely affected by MIH often show extensive disintegration and need complex restorative treatment.2020. William V, Messer LB, Burrow MF. Molar incisor hypomineralization: review and recommendations for clinical management. Pediatr Dent. 2006;28(3):224-32. Consequently, children with MIH usually receive more dental treatment than do unaffected children.2121. Kotsanos N, Kaklamanos EG, Arapostathis K. Treatment management of first permanent molars in children with Molar-Incisor Hypomineralisation. Eur J Paediatr Dent. 2005 Dec;6(4):179-84. Thus, it has been advocated that affected children may be at risk for DFA and DBMPs.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. However, the literature on this subject is controversial.

The knowledge on whether the presence of MIH increases DFA and DBPMs is clinically relevant. Patients with MIH might need a special plan of dental care, focused on a comfortable and calm environment and shorter appointments. Thus, the aim of the present study was to summarize the current available evidence on MIH and the presence of DFA and DBPMs through a systematic review.

Methodology

The purpose of this systematic review was to assess the available evidence on whether children with MIH have more DFA and DBMPs than children without MIH. Its protocol was registered on Prospero (CDR42020203851) and it complied with the PRISMA 2020 statement (http://prisma-statement.org/).

Eligibility criteria

According to the PECO strategy (Population, Exposure, Comparison, and Outcome),2222. Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. J Clin Epidemiol. 2021 Jun;134:178-89. https://doi.org/10.1016/j.jclinepi.2021.03.001
https://doi.org/10.1016/j.jclinepi.2021....
observational studies (cohort, cross-sectional, and case-control studies) that evaluated children and adolescents (P) with MIH (E) compared with individuals without MIH (C), regarding DFA and DBPMs (O), were eligible.

Literature reviews, in vitro studies, case reports, intervention studies, opinion articles, letters to the editor, and theses were not included. Studies based on questionnaires applied to dentists and studies that evaluated patients with special needs or any problem that could interfere with the assessed outcomes were also excluded.

Search

A systematic literature search was performed in MEDLINE/PubMed, Scopus/Elsevier, Web of Science/Clarivate LILACS/VHL, BBO/VHL, Embase/Elsevier, Cochrane Library/Wiley, and APA PsycoINFO databases until March, 2021. Searches in the Open Grey and Google Scholar databases were also performed to assess the gray literature. No restrictions on language or year of publication were imposed to the searches. If any article written in any language other than English, Portuguese, or Spanish was retrieved, a professional translator service would be hired. Reference lists of each selected article were checked manually to find eligible studies not captured by the main search. Expert authors were contacted by e-mail in order to find unpublished or ongoing studies. Alerts were set in all databases to retrieve newly published articles.

The search strategies were created using MeSH terms, entry and free terms in English combined with the Boolean operators AND / OR. The search strategy was firstly developed for PubMed, but adapted for each database. Terms in Portuguese were added to the search strategies for the VHL platform. All these procedures were supervised by an expert librarian (D.F.M.). The search strategies used in each database are provided in Table 1 .

Table 1
Electronic databases and research strategies.

Selection of studies

All the identified records were imported into the EndNote Web software (Thomson Reuters, New York, USA) and duplicates were removed. Titles and abstracts were read by two independent researchers (P.P.G.R. and R.C.J.) to determine the eligibility of the studies. Full articles were retrieved and examined when title and abstract did not provide enough information for a definitive decision. The screened lists of each researcher (P.P.G.R. and R.C.J.) were compared and disagreements were resolved by a third author (V.M.S.).

Data extraction

Two independent reviewers (P.P.G.R. and R.C.J.) extracted the data from the selected studies using an Excel spreadsheet (Microsoft 10). From each study, the following data were extracted: details of the studies (author, year of publication, country, and study design); sample characteristics (number of participants, sex, age), criterion used for the diagnosis of MIH; outcome assessed (DFA, DBPMs); evaluation methods (what instruments were used to measure DFA and DBMPs and who answered them); and results (frequencies of events, prevalence, mean SD, and median). An e-mail was sent to the corresponding author when additional data not found in the articles were necessary.

Quality appraisal

The quality assessment of the selected studies was conducted by two authors separately (PPGR and RCJ) using Newcastle-Ottawa Quality Assessment Scales (NOS).2323. Wells GA, O’Connell D, Petersen J, Welch V, Losos M. The Newcastle-Ottawa Scale (NOS) for assessing the quality of nonrandomized studies in meta-analysis. [place unknow: pubisher unknown]; 2009. Questions and disagreements were answered and solved by a third author (VMS). For cohort and case-control studies, NOS allocates a maximum of nine stars divided into three criteria: selection of the sample (4 stars), comparability (two stars), and outcome/exposure (three stars). For cross-sectional studies, an adapted version of NOS was used,2424. Modesti PA, Reboldi G, Cappuccio FP, Agyemang C, Ramuzzi G, Rapi S, et al. et al. Panethnic differences in blood pressure in Europe: a systematic review and meta-analysis. Plos One, 2016 Jan 25. https://doi.org/10.1371/journal.pone.0147601
https://doi.org/10.1371/journal.pone.014...
with the allocation of a maximum of 10 stars, assigning five stars to selection of the sample.

Representativeness of the population and recruitment of exposed/not exposed patients from the same population were the main factors considered in the quality assessment of sample selection. Assessment of exposure was considered appropriate by trained and calibrated examiners when the diagnosis of hypomineralization was based on specific indices for MIH2525. Weerheijm KL, Duggal M, Mejàre I, Papagiannoulis L, Koch G, Martens LC, et al. Judgement criteria for Molar Incisor Hypomineralisation (MIH) in epidemiologic studies: a summary of the European meeting on MIH held in Athens, 2003. Eur J Paediatr Dent. 2003 SSep;4(3):110-3. , 2626. Ghanim A, Mariño R, Manton DJ. Validity and reproducibility testing of the Molar Incisor Hypomineralisation (MIH) Index. Int J Paediatr Dent. 2019 Jan;29(1):6-13. https://doi.org/10.1111/ipd.12433
https://doi.org/10.1111/ipd.12433...
or developmental defects of enamel.2727. Clarkson J., Mullane DO. A modified DDE Index for use in epidemiological studies of enamel defects. J Dent Res. 1989 Mar;58(3):445-50. https://doi.org/10.1177/00220345890680030201
https://doi.org/10.1177/0022034589068003...
In the comparability domain, the presence of dental caries was considered the most important factor to be controlled and the additional factor was the age range of the sample. Description of independent evaluation of MIH and DFA and/or DBPMs performed by two different examiners was required to regard the assessment of the outcome as independent and blind.

Synthesis methods

Initially, the extracted data were synthesized qualitatively. Demographic characteristics of the samples, outcomes assessed (DFA or DBPMs), and the corresponding assessment methods were evaluated to identify clinical/methodological heterogeneity across the studies. A critical interpretation of the data was carried out to combine results and conclusions. Random-effects meta-analyses were performed to calculate the mean differences (MD) or standardized mean differences (SMD) and corresponding 95% confidence interval (95% CI) of DFA scores between groups with and without MIH. Studies that reported their results as dichotomous data were not included in the quantitative synthesis. Subgroup analyses were performed to explore the variables ‘severity of condition’, ‘age of samples’ and ‘diagnostic criteria for MIH’ as potential sources of heterogeneity. Sensitivity tests were performed to assess the robustness of the analyses. The I2 index was calculated to assess statistical heterogeneity. All analyses were performed using Review Manager 5.4 software.

Certainty of the evidence

The GRADEpro tool2828. Schünemann H, Brozek J, Guyatt G, Oxman A, eds. GRADE Handbook. [cited 2021 Sep 25]. Available from: gradepro.org.
gradepro.org...
was used to rate the certainty of evidence, considering the following items for analysis: risk of bias, inconsistency, indirectness, imprecision, suspicion of publication bias, presence of large effect, dose-effect gradient, and plausible confounders.2828. Schünemann H, Brozek J, Guyatt G, Oxman A, eds. GRADE Handbook. [cited 2021 Sep 25]. Available from: gradepro.org.
gradepro.org...
, 2929. Balshem H, Helfand M, Schünemann HJ, Oxman AD, Kunz R, Brozek J, et al. GRADE guidelines: 3. Rating the quality of evidence. J Clin Epidemiol. 2011 Apr;64(4):401-6. https://doi.org/10.1016/j.jclinepi.2010.07.015
https://doi.org/10.1016/j.jclinepi.2010....
Since no meta-analysis was performed for DBPMs, all the judgments were adapted to qualify the evidence synthesized narratively for this outcome.3030. 3o0. Murad MH, Mustafa RA, Schünemann HJ, Sultan S, Santesso N. Rating the certainty in evidence in the absence of a single estimate of effect. Evid Based Med. 2017 Jun;22(3):85-7. https://doi.org/10.1136/ebmed-2017-110668
https://doi.org/10.1136/ebmed-2017-11066...

Results

Selection of studies

Searches in the databases retrieved 1,411 studies. After removal of duplicates, 520 studies remained, and 12 studies were additionally retrieved from alerts (from March to August 2021). After reading titles and abstracts, 518 reports were excluded for not meeting the inclusion criteria. Thirteen articles were read in full and seven of them were excluded. No eligible study was found through manual search in the reference lists of the included articles. Two records were found in OpenGrey, but both were excluded after reading the title/abstract. From the first 100 matches from Google Scholar, two reports were considered eligible, but only one was selected after reading. Finally, seven studies were included in this review.22. Klingberg G, Broberg AG. Dental fear/anxiety and dental behaviour management problems in children and adolescents: a review of prevalence and concomitant psychological factors. Int J Paediatr Dent. 2007 Nov;17(6):391-406. https://doi.org/10.1111/j.1365-263X.2007.00872.x
https://doi.org/10.1111/j.1365-263X.2007...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

33. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
- 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
The flowchart of the literature search, according to the Prisma 2020 Statement,2222. Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. J Clin Epidemiol. 2021 Jun;134:178-89. https://doi.org/10.1016/j.jclinepi.2021.03.001
https://doi.org/10.1016/j.jclinepi.2021....
is presented in Figure 1 .

Figure 1
Flowchart of the searches and selection of studies according to PRISMA 2020.

Characteristics of the studies

Regarding the data on DFA, all the included studies were considered to be cross-sectional because the participants were selected according to exposure (MIH), and the outcome (DFA) was assessed at a specific point in time. Two studies had been conducted in Sweden,1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
one in Australia,3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
two in Brazil,1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
one in Greece,3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
and one in Turkey.3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
The samples of the studies varied widely in size, from 44 to 2,335 participants, and in age, from 8 to 18 years. All sample studies included male and female participants. Two articles resulted from one study that assessed the same sample in two different periods at an interval of 10 years.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
DFA was assessed at two specific points in time, when children were 8 years old 1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. and later when they were 18 years old,3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
characterizing cross-sectional data. Concerning DBMPs, the data were retrieved retrospectively from the dental records over the previous six years1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. and 10 years,3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
characterizing longitudinal data.

In two studies,1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
MIH was diagnosed using the criteria proposed by EAPD.2525. Weerheijm KL, Duggal M, Mejàre I, Papagiannoulis L, Koch G, Martens LC, et al. Judgement criteria for Molar Incisor Hypomineralisation (MIH) in epidemiologic studies: a summary of the European meeting on MIH held in Athens, 2003. Eur J Paediatr Dent. 2003 SSep;4(3):110-3. Three studies1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
used the DDE criteria,2727. Clarkson J., Mullane DO. A modified DDE Index for use in epidemiological studies of enamel defects. J Dent Res. 1989 Mar;58(3):445-50. https://doi.org/10.1177/00220345890680030201
https://doi.org/10.1177/0022034589068003...
in which the presence of demarcated opacities was regarded as MIH. One study1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
used a newly validated criterion for MIH proposed by Ghanin et al.2626. Ghanim A, Mariño R, Manton DJ. Validity and reproducibility testing of the Molar Incisor Hypomineralisation (MIH) Index. Int J Paediatr Dent. 2019 Jan;29(1):6-13. https://doi.org/10.1111/ipd.12433
https://doi.org/10.1111/ipd.12433...
Another study3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
did not mention which criterion was used for MIH diagnosis.

All included studies1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

33. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
- 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
assessed the association between MIH and DFA and two of them also assessed the association with DBMPs.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
The assessment instruments varied widely. One study3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
used the Modified Child Dental Anxiety Scale – Face (MCDASf)3535. Howard KE, Freeman R. Reliability and validity of a faces version of the Modified Child Dental Anxiety Scale. Int J Paediatr Dent. 2007 Jul;17(4):281-8. https://doi.org/10.1111/j.1365-263X.2007.00830.x
https://doi.org/10.1111/j.1365-263X.2007...
answered by children. One study1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
used the Dental Anxiety Question (DAQ),3636. Neverlien PO. Assessment of a single-item dental anxiety question. Acta Odontol Scand. 1990 Dec;48(6):365-9. https://doi.org/10.3109/00016359009029067
https://doi.org/10.3109/0001635900902906...
which contained a single-item question “Do you think that your child is afraid of going to the dentist?” with four possible answers (“no (1)”, “a little (2)”, “yes, he/she is afraid (3),” and “yes, he/she is very afraid (4)”), responded by parents or guardians. Five studies assessed DFA through the Children’s Fear Survey Schedule – Dental Subscale (CFSS-DS),3737. Beena JP. Dental subscale of children’s fear survey schedule and dental caries prevalence. Eur J Dent. 2013 Apr-Jun;7(2):181-5. https://doi.org/10.4103/1305-7456.110166
https://doi.org/10.4103/1305-7456.110166...
a multi-item self-report scale composed of 15 items, and each can be given five different scores ranging from “not afraid at all (1)” to “very much afraid (5). This instrument was answered by parents1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. in one study and by adolescents3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
in another study. In two studies, children answered the dental subscale.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
In the other study, it was unclear who answered the questions.3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
Two studies also assessed the presence of DBMPs through dental records with disruptive behavior resulting in delay of treatment or rendering treatment impossible.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

The studies used different approaches to classify or measure DFA. Four studies used cut-off points to dichotomize the data. Jalevik1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. and Laureano1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
reported CFSS-DS scores ≥ 38 as presence of DFA. Menoncin1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
considered DAQ scores ≥ 2 as presence of DFA. Özükoç3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
used different cut-off points, rating CFSS-DS scores between 15 and 25 as no dental fear, 26 to 32 as mild dental fear, and 33 to 38 as moderate dental fear. Fear, however, can be kept under control, or fear is borderline and score ≥ 39 indicates severe dental fear. Three studies compared the mean score obtained through the instruments between the groups with and without MIH.3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

33. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
- 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
The data extraction is presented in Table 2 .

Table 2
Data extraction from the included studies.

Methodological quality assessment

The quality assessment according to NOS2323. Wells GA, O’Connell D, Petersen J, Welch V, Losos M. The Newcastle-Ottawa Scale (NOS) for assessing the quality of nonrandomized studies in meta-analysis. [place unknow: pubisher unknown]; 2009. criteria is presented in Table 3 . All the included studies presented methodological issues.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

33. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
- 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
In the selection domain, although all the seven studies recruited exposed and not exposed patients from the same population, in two of them3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
, 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
the sample was not considered representative of the population, and in four of them the sample was relatively small.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
- 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
Only in three studies, exposure was diagnosed according to a specific criterion proposed for MIH.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
In one study,3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
the criteria to define exposure was not clearly explained. Comparability was the most affected domain because five out of seven studies1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
, 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
did not control for caries experience as a confounding factor. Regarding the outcome domain, it was not possible to assure that the assessment of the outcome, both DFA and DBMPs, was independent or blind in any of the studies.

Table 3
Risk of bias of included studies (Newcastle-Ottawa).

Results of individual studies and syntheses

The results of the seven included studies are shown in Table 1 . Overall, most of the studies observed no significant difference in DFA between children with and without MIH.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
, 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
The meta-analysis did not show a significant effect of MIH on the standardized units for the DFA scores (SMD = 0.03; 95%CI: -0.06–0.12; p = 0.53; I2 = 0%; Figure 2 ). Subgroup analyses evidenced no influence of ‘severity of MIH’, ‘age of samples’, and ‘diagnostic criteria for MIH’ on this result. Similarly, sensitivity tests alternating the different reported results according to the MIH severity described by Özükoç3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
or different follow-up periods, as reported by Jalevic and Klingberg,1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
did not alter the significance or direction of the estimated effect. Three studies (two evaluating the same sample in different time periods) considered the severity of MIH in the analysis1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
and only one of them observed significantly higher scores of DFA for MIH children when only the severe cases were considered3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
The meta-analysis including only the results for severe cases of MIH3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
also did not show a significant effect of the condition on DFA scores (MD = 8.68; 95%CI: -8.64–26.00; p = 0.33; I2 = 93%). The studies that evaluated DFA as a dichotomous outcome were considered methodologically heterogeneous, given that in one of them,1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
the data were collected from children and in the other one1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
from the parents; therefore, these data were not combined in a meta-analysis. Only two studies considered caries experience as a potential confounding factor in the analysis.1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

Figure 2
Random-effects meta-analysis on the effect of MIH on DFA scores.

Two articles1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
were obtained from one study that evaluated DBMPs in the same sample at an interval of 10 years. In the first article,1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. the patients were aged 7 to 8 years and DBMPs were significantly more frequent in children with MIH, according to notes retrieved from their dental records. For the second article,3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
the presence of disruptive behavior was checked in the dental records over the previous 10 years and DBMPs were still significantly more frequent in MIH patients.

Certainty of the evidence

The overall certainty of the evidence was very low for both outcomes assessed. All the observational studies presented methodological limitations that could have altered the reported results and seriously affected the evidence. Furthermore, we consider that the indirectness item was also affected because several studies did not use a specific method for the diagnosis of the exposure of interest (MIH). The number of subjects evaluated in relation to DBMPs was insufficient to have conclusive evidence on the matter, thus affecting the imprecision item. The publication bias was considered unsuspected. Because the evidence presented limitations affecting its validity, no item was considered to increase the certainty of the evidence.

Discussion

Patients affected by severe MIH usually need more dental treatment and experience more dental pain and discomfort on repeated occasions than those without it. A longitudinal study suggested these patients are more likely to have DFA and DBMPs.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. Based on that, possible association between the presence of MIH and DFA or DBMPs has been widely cited in the literature. Recently, new studies have found divergent results. The present systematic review was built upon this divergence.

Most studies included in this review did not observe a significant association between MIH and DFA.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
, 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
When similar studies were combined in a meta-analysis, a significant effect of MIH was not observed on the standardized units for the DFA scores. The etiology of DFA might be complex and multifactorial and might differ between individuals from different cultural and social environments, but previous experience with caries and dental pain have been consistently mentioned as relevant factors associated with increased DFA.44. Gao S, Lu J, Li P, Yu D, Zhao W. Prevalence and risk factors of children’s dental anxiety in China: a longitudinal study. BMJ Open. 2021 Apr;11(4):e043647. https://doi.org/10.1136/bmjopen-2020-043647
https://doi.org/10.1136/bmjopen-2020-043...
, 88. Torriani DD, Ferro RL, Bonow ML, Santos IS, Matijasevich A, Barros AJ, et al. Dental caries is associated with dental fear in childhood: findings from a birth cohort study. Caries Res. 2014;48(4):263-70. https://doi.org/10.1159/000356306
https://doi.org/10.1159/000356306...

9. Viswanath D, Krishna AV. Correlation between dental anxiety, Sense of Coherence (SOC) and dental caries in school children from Bangalore North: a cross-sectional study. J Indian Soc Pedod Prev Dent. 2015;33(1):15-8. https://doi.org/10.4103/0970-4388.148962
https://doi.org/10.4103/0970-4388.148962...
- 1010. Silveira ER, Goettems ML, Demarco FF, Azevedo MS. Clinical and individual variables in children’s dental fear: A school - Based investigation. Braz Dent J. 2017;28(3):398-404. https://doi.org/10.1590/0103-6440201601265
https://doi.org/10.1590/0103-64402016012...
In this context, it might be somehow expected that children with MIH present increased DFA when compared with children without MIH, given that it is significantly associated with increased caries indices and higher frequency of restorative procedures.1616. Lygidakis NA, Wong F, Jälevik B, Vierrou AM, Alaluusua S, Espelid I. Best clinical practice guidance for clinicians dealing with children presenting with Molar-Incisor-Hypomineralisation (MIH): an EAPD Policy Document. Eur Arch Paediatr Dent. 2021;22(5)851-860. https://doi.org10.1007/s40368-021-00649-8
https://doi.org10.1007/s40368-021-00649-...
, 3838. Kotsanos N, Kaklamanos EG, Arapostathis K. Treatment management of first permanent molars in children with Molar-Incisor Hypomineralisation. Eur J Paediatr Dent. 2005Dec;179-84. One study observed an increased DFA only in the group with severe MIH.3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
However, the meta-analysis assessing the results of both studies on severe cases of MIH3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
did not show a significant effect of the condition on DFA scores. Further studies should consider the severity of MIH because critical aspects such as treatment need, dental pain, or discomfort may not differ between mild and severe MIH.

Age is another factor that plays a critical role, as it has been shown that DFA tends to decrease over time, being less frequent in older children.22. Klingberg G, Broberg AG. Dental fear/anxiety and dental behaviour management problems in children and adolescents: a review of prevalence and concomitant psychological factors. Int J Paediatr Dent. 2007 Nov;17(6):391-406. https://doi.org/10.1111/j.1365-263X.2007.00872.x
https://doi.org/10.1111/j.1365-263X.2007...
, 44. Gao S, Lu J, Li P, Yu D, Zhao W. Prevalence and risk factors of children’s dental anxiety in China: a longitudinal study. BMJ Open. 2021 Apr;11(4):e043647. https://doi.org/10.1136/bmjopen-2020-043647
https://doi.org/10.1136/bmjopen-2020-043...
For this reason, in the present review, age was an additional confounding factor considered in the quality assessment of the studies. Interestingly, the study that evaluated DFA in the same sample at the ages of 7 to 8 years1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. and later at 18 years3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
observed a significant increase in DFA in MIH children only at the ages of 7 to 8 years. On the other hand, based on the subgroup analyses performed in quantitative syntheses, it should be mentioned that the results were consistent, regardless of the age of the participants.

The presence of DBMPs was evaluated in this review as a secondary outcome. Out of the seven included articles, only two1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
presented data about this outcome assessed in the same sample at a 10-year interval. In both reports, DBMPs were more common in patients with MIH. However, as the data on this outcome were based on notes in the dental records, they might be inaccurate. An appropriate assessment of children’s behavior is important to support dental treatment planning, achieving efficiency and improving patients’ behaviors.3939. Silva JA, Paiva SM, Pereira DM, Maia LC, Primo LG, Fonseca-Gonçalves A. Are behavior rating scales able to identify behavioral changes in preschool children undergoing a dental intervention? A systematic review. Pesq Bras Odontopediatria Clin Integr. 2020:1e5400. https://doi.org/10.1590/pboci.2020.097
https://doi.org/10.1590/pboci.2020.097...
Further studies on the association between DBMPs and MIH through a validated rating scale for behavior evaluation would be desirable to obtain more consistent results.

There is a pool of multi-item and single-item scales to assess DFA in children and adolescents. In the present review, the instruments used to assess DFA and the type of respondents varied widely. Moreover, while some of the studies analyzed the data on DFA as continuous variables,1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...

33. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
- 3434. Kosma I, Kevrekidou A, Boka V, Arapostathis K, Kotsanos N. Molar incisor hypomineralisation (MIH): correlation with dental caries and dental fear. Eur Arch Paediatr Dent. 2016 Apr;17(2):123-9. https://doi.org/10.1007/s40368-016-0221-4
https://doi.org/10.1007/s40368-016-0221-...
other studies used cut-off points to categorize those children and adolescents according to the presence or absence of DFA.1212. Laureano IC, Farias L, Fernandes LH, Alencar CR, Forte FD, Honório DR, et al. Dental fear in children: association with dental caries and molar incisor hypomineralization. Braz Dent J. 2020;31(6):673-9. https://doi.org/10.1590/0103-6440202003880
https://doi.org/10.1590/0103-64402020038...
, 1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...

All the studies included in the present review had methodological limitations. External validity was affected because most of the studies did not have a representative sample or were based on relatively small samples.1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...

32. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
- 3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
Additionally, most of the data were cross-sectional, and then it was not possible to establish a cause-effect relationship between MIH and DFA. In the two reports that assessed DBMPs, using the same sample,1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
it was not possible to guarantee that DBMPs (outcome) had not been present prior to MIH (exposure).

Comparability was a major concern. Caries experience and age were considered the most relevant confounding factors in this review. However, only two studies1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
controlled for both. The presence of dental caries was considered the most important confounding factor because children with dental caries tend to have more pain, more dental treatment needs, and are more susceptible to DFA.88. Torriani DD, Ferro RL, Bonow ML, Santos IS, Matijasevich A, Barros AJ, et al. Dental caries is associated with dental fear in childhood: findings from a birth cohort study. Caries Res. 2014;48(4):263-70. https://doi.org/10.1159/000356306
https://doi.org/10.1159/000356306...
, 4040. Lin CS, Wu SY, Yi CA. Association between Anxiety and pain in dental treatment: a systematic review and meta-analysis. J Dent Res. 2017 Feb;96(2):153-62. https://doi.org/10.1177/0022034516678168
https://doi.org/10.1177/0022034516678168...
, 4141. Folayan MO, Kolawole KA, Onyejaka NK, Agbaje HO, Chukwumah NM, Oyedele TA. General anxiety, dental anxiety, digit sucking, caries and oral hygiene status of children resident in a semi-urban population in Nigeria. BMC Oral Health. 2018 Apr;18(1):66. https://doi.org/10.1186/s12903-018-0529-z
https://doi.org/10.1186/s12903-018-0529-...
However, only two studies controlled for caries in the multivariate analysis testing the association between MIH and DFA.1313. Menoncin BL, Portella PD, Ramos BL, Assunção LR, Souza JF, Menezes JV. Dental anxiety in schoolchildren with molar incisor hypomineralization-A population-based cross-sectional study. Int J Paediatr Dent. 2019 Sep;29(5):615-23. https://doi.org/10.1111/ipd.12503
https://doi.org/10.1111/ipd.12503...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
The age of the participants was considered an additional confounding factor that should be controlled for, given that the first permanent molars erupt around the age of 6 years. MIH molars may fracture due to masticatory efforts over time.4242. Raposo F, Rodrigues ACC, Lia ÉN, Leal SC. Prevalence of hypersensitivity in teeth affected by molar-incisor hypomineralization (MIH). Caries Res. 2019;53(4):424-30. https://doi.org/10.1159/000495848
https://doi.org/10.1159/000495848...
, 4343. Weerheijm KL. Molar incisor hypomineralization (MIH): clinical presentation, aetiology and management. Dent Update. 2004;31(1):9-12. https://doi.org/10.12968/denu.2004.31.1.9
https://doi.org/10.12968/denu.2004.31.1....
The older the children, the longer the tooth will be under masticatory forces, tending to fracture, increasing severity, and possibly causing pain, which eventually might result in more DFA. However, studies that evaluated DFA in children without MIH observed that it tends to decrease with age.44. Gao S, Lu J, Li P, Yu D, Zhao W. Prevalence and risk factors of children’s dental anxiety in China: a longitudinal study. BMJ Open. 2021 Apr;11(4):e043647. https://doi.org/10.1136/bmjopen-2020-043647
https://doi.org/10.1136/bmjopen-2020-043...

A well-trained dentist experienced in dealing with children’s behavior management might be able to reduce DFA and DBMPs in patients, even if they are exposed to repetitive treatments. Therefore, such possibility is a relevant aspect to be considered in this discussion. Preferably, future studies should standardize the way dentists approach these children or should take into consideration the experience and professional qualification background of these dentists.

It should be mentioned that although the results of the syntheses represent the current state of the evidence, they are not conclusive. It is important to emphasize that the results of the meta-analyses should be viewed with caution as they are based on a limited number of studies with methodological issues. In addition, some degree of bias could exist as it was not possible to include all studies in the quantitative synthesis as some reported different measures of effect and were methodologically heterogeneous. This, along with other reasons, resulted in the very low quality of the total body of evidence for both outcomes assessed. Firstly, because all the included studies followed an observational design, which by itself provides a low quality of evidence. Secondly, all studies had methodological limitations that could have influenced the results and seriously affected the evidence. The indirectness item was also affected because three studies1717. Jälevik B, Klingberg GA. Dental treatment, dental fear and behaviour management problems in children with severe enamel hypomineralization of their permanent first molars. Int J Paediatr Dent. 2002 Jan;12(1):24-32. , 3131. Jälevik B, Klingberg G. Treatment outcomes and dental anxiety in 18-year-olds with MIH, comparisons with healthy controls: a longitudinal study. Int J Paediatr Dent. 2012 Mar;22(2):85-91. https://doi.org/10.1111/j.1365-263X.2011.01161.x
https://doi.org/10.1111/j.1365-263X.2011...
, 3232. Arrow P. Dental enamel defects, caries experience and oral health-related quality of life: a cohort study. Aust Dent J. 2017 Jun;62(2):165-72. https://doi.org/10.1111/adj.12449
https://doi.org/10.1111/adj.12449...
evaluated the presence of MIH (exposure of interest) using the DDE index, which is not specific to MIH, and another study3333. Özükoç C. Evaluation of the relationship between MIH severity and dental fear among the children. Med Sci Discov. 2019 Oct;6(10):284-7. https://doi.org/10.36472/msd.v6i10.318
https://doi.org/10.36472/msd.v6i10.318...
did not even mention the criterion used for the diagnosis of MIH. Therefore, the certainty of the evidence was rated down due to the following parameters: study design, methodological issues, risk of bias, and indirectness. Regarding the DBMP outcome, it was not possible to conduct a meta-analysis. The certainty of the evidence was judged following guidelines proposed for cases in which there is no single summary measure of effect.3030. 3o0. Murad MH, Mustafa RA, Schünemann HJ, Sultan S, Santesso N. Rating the certainty in evidence in the absence of a single estimate of effect. Evid Based Med. 2017 Jun;22(3):85-7. https://doi.org/10.1136/ebmed-2017-110668
https://doi.org/10.1136/ebmed-2017-11066...
For DBMPs, the evidence was rated down due to the study design, methodological issues, risk of bias, indirectness, and imprecision.

Conclusion

Based on this systematic review, the evidence suggests that there is no difference in DFA between patients with and without MIH. The two studies reporting on DBMPs observed that behavioral problems were more frequent among children with MIH. These results must be interpreted with caution due to the very low certainty of the evidence. Further studies on the severity of MIH, using validated rating scales for behavior evaluation and controlling for important confounding factors for DFA would be desirable to obtain more consistent results.

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Publication Dates

  • Publication in this collection
    10 July 2023
  • Date of issue
    2023

History

  • Received
    27 Mar 2022
  • Accepted
    10 Oct 2022
  • Reviewed
    25 Nov 2022
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