Quality of Life After Orthognathic Surgery in Patients with Cleft: An Overview of Available Patient-Reported Outcome Measures (2024)

Search

The comprehensive search was performed by a clinical librarian working in theaffiliated medical library. The comprehensive search was performed in thedatabases PubMed/MEDLINE, EMBASE (Ovid), Cochrane Library, and Web of Sciencefrom the inception of the databases until 11 January 2021. The terms used were:orthognathic surgery, orthognathic surgical procedures, maxillofacialabnormalities, dento-maxillary orthopedics AND cleft lip, CP, cleft alveolus,cleft lip alveolus and palate, congenital deformity, dentofacial deformity ANDPRO, surveys and questionnaires, PRO measure, PROM, QOL, life quality,health-related QOL (HRQOL), and psychometrics. The complete search strategiesfor all online databases can be found in Appendix 1. Limits were placed on the search to excludenon-English citations and articles.

Eligibility Criteria

All studies were evaluated by title, keywords, and abstract by 2 reviewers (RPand SW). Discords were resolved by consensus. Full-text reviews of the selectedarticles were independently performed by 2 reviewers (RP and SW). Discords wereagain resolved by consensus. Articles were selected for full review according tothe following a priori eligibility criteria.

Inclusion criteria:

• - Studies evaluating PROM.

• - Studies with descriptions about the evaluation or construct of themeasurement tool.

• - Studies with extensive descriptions of questionnaires used inorthognathic surgery or in patients with dentofacial deformities, orpatients with a cleft, lip (alveolus), and/orpalate.

• - Studies with patient sample sizes were described.

Exclusion criteria:

• - Questionnaires evaluating family conditions only.

Identification and Assessment of Validation of PROMs

Inclusion criteria:

• - Instruments measuring QOL validated for patients with cleftdeformities and for the impact of orthognathic surgery onQOL.

Exclusion criteria:

• - Instruments under construction.

• - Instruments using parents’ or caregivers proxy measures instead ofpatient-reported data.

• - Ad hoc instruments.

• - Instruments are not available in English language.

After identification of instruments measuring QOL, a follow-up search wasperformed by 2 authors RP and SW to check if instruments were both validated forpatients with cleft deformities and for the impact of orthognathic surgery onQOL. Unvalidated instruments were excluded.

Quality Assessment

The methodological quality of the studies, describing the PROMs suitable forquality assessment, were evaluated with the consensus-based standards for theselection of health status measurement instruments (COSMIN) risk of biaschecklist (Mokkink et al.,2018; Terwee etal., 2018; Prinsen et al., 2018). The COSMIN checklist contains 10 boxes toassess the included studies’ methodological quality of the measurementproperties, using a 4-point scale ranging from very good, adequate, doubtfultill inadequate. Measurement properties not possible to evaluate were rated notapplicable. A detailed description of use, evaluation, and scores of the COSMINrisk of bias checklist can be found in the COSMIN methodology for systematicreviews of patient-reported outcome measures (PROMs) user manual (Mokkink et al., 2018;Prinsen et al.,2018; Terwee etal., 2018).

PROMs measuring the impact of orthognathic surgery on QOL in patients with cleftdeformities were assessed using the quality criteria proposed for measurementproperties of health status questionnaires (Terwee et al., 2007). Positive,intermediate, negative or no information available ratings can be given to themeasurement properties: (1) content validity, (2) internal consistency, (3)criterion validity, (4) construct validity, (5) reproducibility, (6)responsiveness, (7) floor and ceiling effects, and (8) interpretability. Thedescription of the defined quality criteria and rating system is detaileddescribed (Terwee et al.,2007).

Table 1.

Identified Instruments Measuring Quality of Life.

Risk of Bias Checklist

The CLEFT-Q has been identified as a validated PROM measuring the impact oforthognathic surgery on QOL in a patient with a cleft lip (alveolus) and/orpalate and is described in 4 studies: Wong Riff et al. (2017), Klassen et al. (2018),and Harrison et al.(2019). It consists of 12 different scales measuring appearance (ofthe nose, teeth, lips, jaws, cleft lip scar, and the face), speech function(social, school, speech distress, psychological), and HRQOL. Lower scale scoresare associated with facial appearance dissatisfaction, speech problems, and aneed for future treatment regarding cleft-related problems (Wong Riff et al.,2017; Klassen et al.,2018). Three studies, Klassen et al. (2018), and Harrison et al. (2019),extensively describe the construct and validation of the CLEFT-Q.

Results of the COSMIN risk of bias checklist of the 4 included studies show thatthe methodological quality of the studies scores is “very good.” Wong Riff et al.(2017) described only the study protocol of the development of theCLEFT-Q (Wong Riff et al.,2017) therefore no scores were given for this study (Table 2).

Quality Assessment of PROMs

Only 1 PROM, the CLEFT-Q, was assessed with the quality criteria proposed formeasurement properties of health status questionnaires (Terwee et al., 2007). The qualitycriteria for the 8 attributes of instrument properties: (1) content validity,(2) internal consistency, (3) criterion validity, (4) construct validity, (5)reproducibility, (6) responsiveness, (7) floor and ceiling effects, and (8)interpretability were evaluated and are summarized in Table 3 (Terwee et al., 2012).

Specification:

1. The content validity is described extensively. Target population,concepts, and item selection are discussed, therefore contentvalidity is considered positive (Klassen et al.,2018).

2. The internal consistency shows Cronbach α values ranging from .89 to.96 (Cronbach's alpha is considered good between .70 and .95).Regarding factor analyses, the sample size of the CLEFT-Q was >7times the number of items, and the sample size was >100 (Klassen et al.,2018). Therefore, internal consistency is consideredpositive.

3. Currently, there is no information available to evaluate thecriterion validity of the CLEFT-Q. In the study protocol publishedby the developers, the criterion validity is planned to be furtherelaborated in phase 3 of the construction of the CLEFT-Q (Wong Riff et al.,2017).

4. Construct validity of the CLEFT-Q is assessed by a priori hypotheses.Rasch analysis provided evidence of reliability and validity for 12of 13 scales (Klassen et al., 2018). Construct validity of the CLEFT-Qis considered positive because specific hypotheses were formulatedand at least 75% of the results are in accordance with thesehypotheses.

5. Reproducibility can be divided into (5a) agreement and (5b)reliability. No information was found for the agreement. Thereliability Person separation index values were ≥0.85 for 10/12scales (intraclass correlation coefficient [ICC] or ICC or weightedKappa >0.70 is considered good) (Klassen et al., 2018). Thereliability of the CLEFT-Q is therefore consideredpositive.

6. The responsiveness is also planned to be further elaborated in phase3 of the construction of the CLEFT-Q (Wong Riff et al., 2017;Harrison etal., 2019). At the moment there is no informationavailable for this attribute.

7. In all 4 articles, floor or ceiling effects are not mentioned,therefore assessment is not applicable for floor or ceilingeffects.

8. For the last attribute, the interpretability, the mean and standarddeviation (SD) scores are defined in 4 relevant subgroups ofpatients (Harrison et al., 2019). The definition of the minimalimportant change (MIC) is scheduled in phase 3 of the CLEFT-Qconstruction (Wong Riff et al., 2017). With the criteria “mean and SDscores presented off at least four relevant subgroups of patientsand no MIC defined” (Terwee et al., 2007), theinterpretability of the CLEFT-Q is assessed as“intermediate.”

Results Compared to Previous Research

In previously conducted searches for instruments measuring the QOL in patientswith a cleft in general, 5 and 6 validated instruments were found by Ecksteinand Klassen, respectively (Eckstein et al., 2011; Klassen et al., 2012). Since these searches have beenperformed until 2011, more cleft-specific PROMs have subsequently beenconstructed over time, resulting in the CLEFT-Q and 13 other validatedinstruments (visual analog scale [VAS], child oral health impact profileshort-form health survey-19 [COHIP-SF19], child perception questionnaire 8-11[CPQ 8-11], Child Perception Questionnaire 11-14 [CPQ 11-14], VPI effects onlife outcomes [VELO], pediatric QOL inventory [PEDSQL], patient-reportedoutcomes measurement information system [PROMIS], QOL in adolescent with cleftassessment [QLACA], pediatric voice-related QOL [PVRQOL], youth QOL instrumentfacial differences [YQOL FD], youth QOL instrument craniofacial surgery [YQOLCS], MOHRQOL, and the cleft hearing, appearance and speech questionnaire[CHASQ]; Table 1).In addition, several cleft-parents or caregivers-report instruments and cleftinstruments under construction were identified in the present search, but didnot meet the inclusion criteria of this study. In a previously conducted searchby Zamboni et al.(2019), regarding instruments measuring the impact of orthognathicsurgery on QOL 7 PROMs were found. Most of those questionnaires were notproperly validated, and only the orthognathic QOL questionnaire (OQLQ) met theinclusion criteria of the present study.

PROMs such as the oral health impact profile 14 (OHIP-14), short-form healthsurvey-36 (SF-36), World Health Organization QOL-BREF (WHOQOL-BREF), sence ofcoherence questionnaire-29, VAS, and patient atisfaction questionnaire have beenused in single or multiple studies investigating the impact of orthognathicsurgery on QOL (Zamboni etal., 2019). These PROMs have not been validated for measuringorthognathic QOL in patients with a cleft.

CLEFT-Q

The only valid PROM found in this search, the CLEFT-Q, scores mainly “very good”after evaluating the methodological quality with the COSMIN checklist (Terwee et al., 2012).Quality assessment of the CLEFT-Q demonstrates a positive score in contentvalidity, internal consistency, construct validity, and reliability and anintermediate score in interpretability. There is no information yet availablefor criterion validity, agreement, responsiveness, and floor- and ceilingeffects. The proposed quality criteria by Terwee et al. (2007), consider contentvalidity as 1 of the most important measurement properties. They state that whenthe content validity has shown to be adequate, the questionnaire is allowed tobe used. The CLEFT-Q scores positive in content validity and several othercriteria and is therefore considered as immediately usable for clinicians (Klassen et al.,2018).

The CLEFT-Q has been translated into multiple languages and scores very high incross-cultural validity (Tsangaris et al., 2018). Non-English instruments were outside thedesign of this study, and thus not included in the assessment.

Limitations of This Study

The majority of cleft-orientated studies or constructed cleft-specific PROMsfocus on patients aged 0 to 21 years. Due to a large number of young patients,several PROMs make use of the reports of parents or caregivers proxy (Klassen et al., 2012).It appears that there is a good concordance between the reports of parents orcaregivers proxy and children in relationship to children's oral health (Wilson-Genderson et al.,2007; Clawson etal., 2013). With an average age of 17 years or more during theorthognathic surgical intervention (Yamaguchi et al., 2016), parents orcaregivers proxy focused PROMs are considered not contributive.

Non-English articles and instruments were excluded in the present study,therefore possible PROMs could have been overlooked. The researchers conductedfollow-up searches for every PROM identified as thoroughly as possible andprevious systematic reviews have been scanned for PROMs, but cannot completelyrule out an incomplete literature search.

No specific orthognathic procedures have been determined for inclusion andexclusion a priori. All articles describing skeletal corrections of the jaws andface have been included. In the article describing further construct validationof the CLEFT-Q, only “Jaw surgery” has been described regarding orthognathicsurgery (Harrison et al.,2019). It is a suggestion for future studies to specifically describethe orthognathic procedure executed in patients.

sj-docx-1-cpc-10.1177_10556656211067120 - Supplemental material forQuality of Life After Orthognathic Surgery in Patients with Cleft: AnOverview of Available Patient-Reported Outcome Measures:

Supplemental material, sj-docx-1-cpc-10.1177_10556656211067120 for Quality ofLife After Orthognathic Surgery in Patients with Cleft: An Overview of AvailablePatient-Reported Outcome Measures by Roan L. M. Ploumen, Samuel H. Willemse,Ronald E. G. Jonkman, Jitske W. Nolte and Alfred G. Becking in The Cleft PalateCraniofacial Journal

The authors declared no potential conflicts of interest with respect to theresearch, authorship, and/or publication of this article.

Funding: The authors received no financial support for the research, authorship, and/orpublication of this article.

ORCID iDs: Roan L. M. Ploumen https://orcid.org/0000-0003-4012-2291

Samuel H. Willemse https://orcid.org/0000-0002-4527-0528

Ronald E. G. Jonkman https://orcid.org/0000-0002-5905-3639

Jitske W. Nolte https://orcid.org/0000-0002-1570-6263

Alfred G. Becking https://orcid.org/0000-0002-6371-4671

Supplemental Material: Supplemental material for this article is available online.

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