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Mira Nassar

Fifth year, Group 12

Evaluation of Oral Health-Related Quality of

Life of Patients with Cleft Lip and/or Palate

Based on the COHIP Index

A Systemic Review

Master Thesis

Supervisor:

D.D.S, PhD, Prof. Arūnas Vasiliauskas

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FINAL MASTER‘S THESIS IS CONDUCTED

AT THE DEPARTMENT OF ORTHODONTICS

STATEMENT OF THESIS ORIGINALITY

I confirm that the submitted Final Master‘s Thesis: “Evaluation of Oral Health-Related Quality of Life of Patients with Cleft Lip and/or Palate Based on the COHIP Index. A Systemic Review” 1. Is done by myself.

2. Has not been used at another university in Lithuania or abroad.

3. I did not used any additional sources that are not listed in the Thesis, and I provide a complete list of references.

I confirm by e-mail, and the work will be signed after the end of the quarantine and emergency situation due to the COVID-19 pandemic in the republic of Lithuania.

(date) (autthor‘s full name) (signature)

Mira Nassar

CONCLUSION OF FINAL MASTER‘S THESIS ACADEMIC SUPERVISOR

ON THE DEFENSE OF THE THESIS

I confirm by e-mail, and the work will be signed after the end of the quarantine and emergency situation due to the COVID-19 pandemic in the republic of Lithuania.

(date) (author‘s full name) (signature)

Arūnas Vasiliauskas

FINAL MASTER‘S THESIS IS APPROVED AT THE DEPARTMENT

I confirm by e-mail, and the work will be signed after the end of the quarantine and emergency situation due to the COVID-19 pandemic in the republic of Lithuania.

(date of approval) (name of the Department and full name of the Head of the Department) (signature) Department of Orthodontics

Final Master‘s Thesis reviewer

I confirm by e-mail, and the work will be signed after the end of the quarantine and emergency situation due to the COVID-19 pandemic in the republic of Lithuania.

(full name) (signature)

Evaluation of Final Master‘s Thesis Defense Board:

I confirm by e-mail, and the work will be signed after the end of the quarantine and emergency situation due to the COVID-19 pandemic in the republic of Lithuania.

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LITHUANIAN UNIVERSITY OF HEALTH SCIENCES MEDICAL ACADEMY

FACULTY OF ODONTOLOGY CLINIC OF ORTHODONTICS

Evaluation of Oral Health Related Quality of Life of Patients with Cleft Lip and/or Palate Based on the COHIP Index

A Systemic Review

Master Thesis

The thesis was done

by student ……… Supervisor……… (Signature) (signature) …Mira Nassar, Fifth year, OF12… D.D.S., PhD, prof., Arūnas Vasiliauskas

(name, surname, year, group) (degree, name, surname) …30-04-2020…. ………20…. (day/month) (day/month)

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EVALUATION

TABLE

OF

THE

MASTER’S

THESIS

OF THE TYPE OF SYSTEMIC REVIEW OF SCIENTIFIC LITERATURE

Evaluation: ... Reviewer: ...

(Scientific degree. Name, Surname) Reviewing date: ... No. MT parts MT evaluation aspects Compliance with MT requirements and evaluation Yes Partially No 1 Summary (0.5 point)

Is summary informative and in compliance with the

thesis content and requirements? 0.3 0.1 0

2 Are keywords in compliance with the thesis

essence? 0.2 0.1 0

3 Introduction,

aim and tasks (1 point)

Are the novelty, relevance and significance of the

work justified in the introduction of the thesis? 0.4 0.2 0

4 Are the problem, hypothesis, aim and tasks formed

clearly and properly? 0.4 0.2 0

5 Are the aim and tasks interrelated? 0.2 0.1 0

6 Selection criteria of the studies, search methods and strategy (3.4 points)

Is the protocol of systemic review present? 0.6 0.3 0

7

Were the eligibility criteria of articles for the selected protocol determined (e.g., year, language, publication condition, etc.)

0.4 0.2 0

8

Are all the information sources (databases with dates of coverage, contact with study authors to identify additional studies) described and are the last search day indicated?

0.2 0.1 0

9

Is the electronic search strategy described in such a way that it could be repeated (year of search, the last search day; keywords and their combinations; number of found and selected articles according to the combinations of keywords)?

0.4 0.1 0

10 Is the selection process of studies (screening, eligibility, included in systemic review or, if applicable, included in the meta-analysis) described?

0.4 0.2 0

11

Is the data extraction method from the articles (types of investigations, participants,

interventions, analysed factors, indexes) described?

0.4 0.2 0

12

Are all the variables (for which data were sought and any assumptions and simplifications made) listed and defined?

0.4 0.2 0

13

Are the methods, which were used to evaluate the risk of bias of individual studies and how this

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Information is to be used in data synthesis, described?

14 Were the principal summary measures (risk ratio,

difference in means) stated? 0.4 0.2 0

15 Systemization and analysis of data (2.2 points)

Is the number of studies screened: included upon assessment for eligibility and excluded upon giving the reasons in each stage of exclusion presented?

0.6 0.3 0

16 Are the characteristics of studies presented in the included articles, according to which the data were extracted (e.g., study size, follow-up period, type of respondents) presented?

0.6 0.3 0

17 Are the evaluations of beneficial or harmful outcomes for each study presented? (a) simple summary data for each intervention group; b) effect estimates and confidence intervals)

0.4 0.2 0

18

Are the extracted and systemized data from studies presented in the tables according to individual tasks? 0.6 0.3 0 19 Discussion (1.4 points)

Are the main findings summarized and is their

relevance indicated? 0.4 0.2 0

20 Are the limitations of the performed systemic

review discussed? 0.4 0.2 0

21 Does author present the interpretation of the

results? 0.4 0.2 0

22

Conclusions (0.5 points)

Do the conclusions reflect the topic, aim and tasks

of the Master’s thesis? 0.2 0.1 0

23 Are the conclusions based on the analysed

material?

0.2 0.1 0

24 Are the conclusions clear and laconic? 0.1 0.1 0

25

References (1 point)

Is the references list formed according to the

requirements? 0.4 0.2 0

26

Are the links of the references to the text correct? Are the literature sources cited correctly and precisely?

0.2 0.1 0

27 Is the scientific level of references suitable for

Master’s thesis? 0.2 0.1 0

28

Do the cited sources not older than 10 years old form at least 70% of sources, and the not older than 5 years – at least 40%?

0.2 0.1 0

Additional sections, which may increase the collected number of points

29 Annexes Do the presented annexes help to understand the

analysed topic? +0.2 +0.1 0

30

Practical

recommendations Are the practical recommendations suggested and

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31

Were additional methods of data analysis and their results used and described (sensitivity analyses, meta-regression)?

+1 +0.5 0

32

Was meta-analysis applied? Are the selected statistical methods indicated? Are the results of each meta-analysis presented?

+2 +1 0

General requirements, non-compliance with which reduce the number of points

33 General requirements

Is the thesis volume sufficient (excluding annexes)?

15-20 pages (-2 points)

<15pages (-5points)

34 Is the thesis volume increased

artificially? -2 points -1 point

35 Does the thesis structure satisfy the requirements of Master’s thesis? -1 point -2 points 36 Is the thesis written in correct language,

scientifically, logically and laconically? -0.5 point -1 points

37 Are there any grammatical, style or

computer literacy-related mistakes? -2 points -1 points 38

Is text consistent, integral, and are the volumes of its structural parts

balanced?

-0.2 point -0.5 points

39 Amount of plagiarism in the thesis. >20% (not

evaluated)

40 Is the content (names of sections and

sub- sections and enumeration of pages) in compliance with the thesis structure and aims?

-0.2 point -0.5 points

41 Are the names of the thesis parts in

compliance with the text? Are the titles of sections and sub-sections

distinguished logically and correctly?

-0.2 point -0.5 points

42 Are there explanations of the key terms

and abbreviations (if needed)? -0.2 point -0.5 points

43

Is the quality of the thesis typography (quality of printing, visual aids, binding) good?

-0.2 point -0.5 points

*In total (maximum 10 points):

*

Remark: the amount of collected points may exceed 10 points.

Reviewer’s comments: ___________________________________________________________ ______________________________________________________________________________ ________________________________________________________________________________ ____________________________________________________________________

Reviewer’s name and surname Reviewer’s signature

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TABLE OF CONTENTS

ABSTRACT ... 8 1. INTRODUCTION ... 9 1.1 Aim ... 10 1.2 Tasks ... 10 1.3 Hypothesis: ... 10 ABBREVIATIONS ... 11 COHIP DESCRIPTION ... 12

2. SEARCH METHODS AND STRATEGY ... 13

2.1 Focus Question ... 13

2.2 Types of publications... 13

2.3 Types of studies ... 13

2.4 Population ... 13

2.5 Data collection ... 14

2.6 Literature search and screening ... 14

2.7 Selection of studies ... 15

2.8 Inclusion and exclusion criteria ... 15

2.9 Assessment of risk of bias ... 17

Identification... 18

3. SYSTEMIZATION AND ANALYSIS OF DATA ... 18

3.1 Study Selection ... 18

3.2. Evaluation of the differences between children and their parents ... 19

3.3 Evaluation of the differences between different age groups according to the COHIP index .. 21

3.4 Evaluation of the differences between different genders according to the COHIP index ... 23

3.5 Evaluation of the differences between different cleft types according to the COHIP index ... 25

4. DISCUSSION ... 27

5. CONCLUSIONS ... 31

6. PRACTICAL RECOMMENDATIONS... 32

7. REFERENCES ... 33

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8 Evaluation of Oral Health-Related Quality of Life of Patients with Cleft Lip and/or Palate Based

on the COHIP Index A Systemic Review

ABSTRACT

Objectives: To determine if there’s a discrepancy in cleft lip and/or palate (CL/P) patients’ oral

health-related quality of life (OHRQoL) between different genders, age groups, and cleft types. To identify if the children’s responses to their quality of life differ from those of their caregivers’.

Materials and methods: Ebsco Publishing, Cochrane, and PubMed/Medline databases were

electronically searched to retrieve results. Articles included studies in English on patients aged 7-19 years. The COHIP index evaluated the child's oral health, functional well-being, social-emotional well-being, school environment, self-image, and the child's overall OHRQoL.

Results: The primary search produced 548 articles, and with duplicate removal, after the

application of inclusion and exclusion criteria, 8 articles were eligible for this review. Parents ranked higher than their children on the "Oral Health" sub-scale (40.52 vs. 24.8) and lower on the "Functional well-being" sub-scale (16.1 vs. 35.6). Older patients were affected more than the younger, specifically on the "Emotional well-being" (25.3 vs.32.36) and "Oral Health" (11.7 vs. 40.04) sub-scales. Females had greater OHRQoL than males except for the “Oral Health” status. Bilateral cleft lip and palate patients (BCLP) had one of the lowest sub-scales and overall COHIP scores.

Conclusion: Younger patients with CL/P had better scores of OHRQoL on the emotional

well-being and oral health sub-scales, mainly. Females had a better OHRQoL than males except for oral health. Children’s sentiments varied from those of their parents, particularly on the oral and functional well-being sub-scales. Children with BCLP were the most affected than any other type of cleft.

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1. INTRODUCTION

Orofacial clefts are among the most common congenital deformities, with a prevalence estimated 1 per 700 live births [1]. The genetic existence is an important etiological factor for cleft lip and/or palate (CL/P), but also environmental factors may play a role in the occurrence [2].

Oral clefts are generally classified as cleft lip, with or without a cleft palate or cleft palate only. A complete cleft lip (CL) is the defect that covers the whole upper lip, while when it is incomplete, it involves only part of the lip. Deformity of the CL develops in the first 6–8 weeks of pregnancy and is usually considered being caused by fusion failure between the maxillary and median nasal processes. A palatal cleft (CP) results from the failure of the fusion of the palatal shelves of the maxillary processes. The degree of the palatal cleft can vary from complete, extending bilaterally on each side of the premaxilla, to a simple bifid uvula.

Clefts affect the patient's well-being, including functional, for example, hyper-nasality, gulping, hearing, and morphological issues, like, deformed nose and mouth asymmetry, which affect the psychosocial status and public activity. Clinical issues related to clefts credit to a velopharyngeal disability, supernumerary, missing, and poorly aligned teeth, constricted maxillary arch, etc. [3]

Children with orofacial clefts experience multiple treatment procedures from birth to maturity. The success of the medical treatment of these children is not just characterized by closure or correction of the cleft. Yet, besides, it reaches out to the support or improvement of the child’s quality of life (QoL) after therapy [4].

Health-related quality of life (HRQoL) enables health professionals to assess how the disease, with its inconveniences and treatments, influences the individual’s life.

Oral Health-related quality of life (OHRQoL) is a term that is utilized to depict the impact of patients' oral health condition on their quality of life. Topolski et al., [5] and several other studies have found that children with visible facial defects have more deficient measures of quality of life compared to healthy children. Infants with isolated cleft palate gradually improve the quality of life scores as they approach adolescence. However, quality of life scores for children with cleft lips decline when they reach puberty as their peers become more critical of their acceptance [6].

There is a range of instruments used to measure the quality of life, such as the Quality of Life Assessment Instrument of the World Health Organization (WHOQOL), Oral Health Impact Profile (OHIP), Child Oral Health Impact Profile (COHIP) and Youth Quality of Life

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10 Instrument-Craniofacial Surgery (YQOL-CS), which seeks to assess the effect of surgery on the life of an individual from their own point of view [7] (Annex 1).

Broder and Wilson developed in 2007 the Child Oral Health Impact Profile (COHIP) to assess the OHRQoL in younger people [8] (Annex 2). This instrument helps to monitor the oral, functional, and social-emotional well-being, and also the school environment, self-image, and the overall OHRQoL of the child. They achieve this through a questionnaire given to both the patients and their caregivers. The resultant scale provided a high level of internal consistency and excellent test-retest reliability, leading to a valuable tool for evaluating oral health-related quality of life in CL/P children [9].

However, little work has been carried out using the COHIP instrument for studying the OHRQoL of children with orofacial clefts on various aspects. This systematic review was performed on articles investigating the quality of life in children with cleft lip and/or palate based on the measurement index COHIP of OHRQoL. This research aimed to evaluate the effect that orofacial clefts have on children's quality of life.

1.1 Aim

This study aims to assess the impact of orofacial clefts on the quality of life of children between the ages of 7 and 19 years.

1.2 Tasks

• Determine if responses of children on their quality of life differ from their caregivers. • Determine if the quality of life is different according to age groups.

• Determine if there is a difference in the quality of life between genders.

• Determine if there is a difference in the quality of life of children with different types of clefts.

1.3 Hypothesis:

This systematic review hypothesizes that children's quality of life differs between types of clefts, different age groups, genders, and also their response to OHRQoL differs from the opinion of their caregivers.

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ABBREVIATIONS

• OHRQoL - Oral Health-Related Quality of Life • HRQoL – Health-Related Quality of Life • COHIP - Child Oral Health Impact Profile • CL - Cleft lip

• CP - Cleft palate

• UCLP - Unilateral cleft lip, palate • BCLP - Bilateral cleft lip, palate • QoL - Quality of life

• CL/P - Cleft lip and/or palate

• CL(A) - Cleft lip with or without alveolus • PRO – Patient-reported outcome

• COHIP-SF - The Child Oral Health Impact-Short Form • AXIS - Appraisal tool for Cross-Sectional Studies

• WHOQOL - Quality of Life Assessment Instrument of the World Health Organization • OHIP - Oral Health Impact Profile

• YQOL-CS -Youth Quality of Life Instrument-Craniofacial Surgery • CA - Critical Appraisal

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COHIP DESCRIPTION

The Child Oral Health Impact Profile (COHIP) consists of two parallel questionnaires, one for the child and one for the caregiver, regarding the child's cleft experience.

This study includes children with a history of CL/P with an age range from 8 to 15 years.

The survey has 34 things in five areas: oral, functional, social-emotional well-being, school

condition, and mental self-view. Elements were scored on a five-point scale (from 1 = never to

5 = practically always), with another extra response choice of 'I don't know.' The scores on

contrarily planned things were turned around before scoring. Higher COHIP scores replicate progressively positive OHRQoL, whereas lower scores replicate lower OHRQoL. Sub-scale scores were determined by adding the reactions of the things explicit to the scale, and

sub-scales were added for the general OHRQoL scores [8-10].

In the articles used in this study, some adjustments have been made to this instrument. The Child Oral Health Impact-Short Form (COHIP-SF) used by Agnew et al. [11] is a significantly shorter questionnaire that decreases the burden of response and facilitates faster, more effective implementation in clinical practice and research. The COHIP-SF was found to have strong psychometric properties comparable to the original COHIP for children and adolescents to test OHRQoL.

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2. SEARCH METHODS AND STRATEGY

Bioethics Approval Code- BEC-OF(U)-117 2.1 Focus Question

The focus question was developed according to the problem, intervention, comparison, and outcome study design. (PICO) is presented in Table 1. [12].

Table 1. PICO table

Components Description

Population (P) Patients of ages 7-19 years after the treatment of cleft lip/palate.

Intervention (I) COHIP index after cleft lip/palate treatment procedures.

Comparison (C) Comparison of the quality of life perception between children and

their caregivers, different age groups, genders, and different types of clefts.

Outcome (O) Children’s quality of life and their response to OHRQoL depends

on different types of clefts, genders, age, and their caregivers.

Focus Question Do clefts affect the oral health-related quality of life of children

when comparing within different types of clefts, different age groups, genders, and their caregivers?

2.2 Types of publications

The systematic review included studies on humans published in the English language.

2.3 Types of studies

The systematic review included all human cross-sectional and cohort studies published between 2008-2020, that reported the quality of life of children with cleft lip and palates.

2.4 Population

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2.5 Data collection

Information and articles gathered from Ebsco Publishing, Cochrane, and PubMed/Medline databases.

2.6 Literature search and screening

To detect the appropriate studies, a thorough electronic search was carried out according to PRISMA guidelines within Ebsco Publishing, Cochrane and PubMed/Medline databases using different combinations of the following keywords: (Table 2)

“Cleft of lip and palate” AND “Quality of life”

“cleft lip, palate” AND “oral health-related quality of life”

“cleft lip, palate” AND “Child oral health impact profile”

Also, a manual search was performed to find additional relevant articles and references.

Table 2: Summary of keyword combination results

Search dates Keywords Results

9/9/2019 - PubMed quality of life with cleft lips and palates

252

15/9/2019 - PubMed oral health-related quality of life, cleft lip, palate

91

23/9/2019 - PubMed child oral health impact profile, cleft lip, palate

26

27/10/2109 - Cochrane quality of life with cleft lip and palate

4 27/11/2019- Ebsco cleft of lip and palate, quality

of life

362

25/1/2020- Ebsco (5 years) cleft of lip and palate, quality of life

239

20/3/2020 -PubMed – last search date

(quality of life) AND cleft

lip, cleft palate

261

20/3/2020 – Ebsco (2008-2020) – last search date

(quality of life) AND cleft

lip, cleft palate

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2.7 Selection of studies

Searched articles were clarified according to inclusion and exclusion criteria. Primarily titles and abstracts were obtained for all the studies that were considered adequate for inclusion in this systematic review (Figure 1).

2.8 Inclusion and exclusion criteria Inclusion criteria for the assortment were:

• Studies done on children between the ages of 7-19 years after cleft lip and palate treatment

• Articles written in the English language • Clinical studies performed on humans • Using the COHIP instrument

Exclusion criteria for the assortment were :

• Instruments other than COHIP • Studies done on older patients • Studies not in the English language • Systematic reviews

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Figure 1. Prisma Flow Chart

Additional records identified through other sources

(n =0)

Records after duplicates removed (n = 330)

Records screened (n = 330)

)

Records excluded: non-relevant title and abstract,

different languages, instruments, and tasks

(n =240)

Full-text articles assessed for eligibility

(n = 90)

Full-text articles excluded, with reasons: different instruments, different age

groups, different tasks (n =82)

Studies included in the qualitative synthesis (n =8) Identification Screening Eligibility Included

Records identified through database searching (PubMed, Ebsco,

Cochrane) (n =548)

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2.9 Assessment of risk of bias

The appraisal tool for Cross-Sectional Studies (AXIS) was used for evaluation of bias. (Annex 3) This evaluation instrument was created for use in assessing observational cross-sectional examinations. [13]. (Table 3)

Table 3: Risk of bias

*Refer to Annex 3 for questions 1-20 (The examination instrument has zones to record a "yes," "no," or "don't know" answer for each question.)

Question * 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 Article Agnew et al. [11]

Yes Yes No Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes Yes I don’t

know Yes

Konan et al. [14]

Yes Yes Yes Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes No I don’t

know Yes

Bos et al. [15]

Yes Yes Yes Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

Yes Yes Yes Yes No I don’t

know No

Nolte et al. [16]

Yes Yes Yes Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes Yes I don’t

know Yes

Ward et al. [17]

Yes Yes Yes Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes Yes I don’t

know Yes

Abebe et al. [18]

Yes Yes Yes Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes Yes I don’t

know Yes

Eslami et al. [19]

Yes Yes No Yes Yes Yes No Yes Yes Yes Yes Yes I don’t

know

No Yes Yes Yes No I don’t

know Yes

Broder et al. [20]

Yes Yes No Yes Yes Yes No Yes Yes No Yes Yes I don’t

know

No Yes Yes Yes No I don’t

know No

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3. SYSTEMIZATION AND ANALYSIS OF DATA

3.1 Study Selection

In total, the primary search produced 548 articles, 330 of which were duplicates. After a primary review of the titles, abstracts, and according to exclusion and inclusion criteria, 240 were omitted. Due to limited access and non-relevant content, 82 articles were omitted when reviewing full-text posts. Finally, 8 eligible studies involving patients aged between 7 and 19 years were enlisted in this systematic review. The systematization of data is presented in Table 4.

Table 4. Systemization of data and characteristics of studies

Study Sample Size

Country Age groups Gender Caregivers Cleft Types Type of Study Evaluation Instrument Agnew et al. [11] 2017 222 Australia (7-10) 109 (11-14)63 (15-18)50 133Males 89Females - 91UCLP 45CP 36BCLP 22CL Cross -sectional (proxy report) COHIP-SF Konan et al. [14] 2015 140 Thailand (8-11)65 (12-15)75 70Males 70Females 140 30CLA 79UCLP 29BCLP 2CP Cross -sectional COHIP Bos et al. [15] 2011 122 Netherland s (8-11)64 (12-15)58 69Males 53Females 122 33CP 38CLA 32UCLP 19BCLP Cross -sectional COHIP Dutch version Nolte et al. [16] 2019 170 Netherland s (8-18) 94Males 76Females 170 50CP 42CL(A) 54UCLP 24BCLP Follow up study Prospectiv e blinded questionna ires. COHIP Dutch version Ward et al.[17] 2013 75 USA (8-10)22 (11-14) 35 (15-18)18 48Males 27Females 75 - Cross -sectional COHIP Abebe et al.[18] 2018 41 Ethiopia (8-17) 21Males 20Females - 24UCLP 9BCLP 2BCL 3UCL 3CP Cross -sectional COHIP Eslami et al. [19] 2013 50 Iran (8-15) 24Males= 26Females - 26UCLP 24BCLP Cross -sectional COHIP Broder et al.[20] 2012 839 USA (7-19) 55%Males 45%Femal es - - Cross -sectional COHIP

COHIP-Child Oral Health Impact Profile; CL(A)-cleft lip/alveolus; UCLP-unilateral cleft lip, palate; CP-cleft

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3.2. Evaluation of the differences between children and their parents

Overall and sub-scale COHIP scores and differences between children and parents are presented in Table 5.

In the study conducted by Konan et al. in 2015 [14], parents had higher self-image impressions of their children than the children themselves (21.19 vs. 19.75), regardless, of only compelled extents of assortment in the general perspective of their children's OHRQoL by patients and parents. Sub-scales scores of parents were more than patients’ scores on overall COHIP, "Oral symptom," "School environment," and "Self-image." From their outcome of the current study, the expectations of parents regarding their child's OHRQoL did not differ from the child itself. Bos et al. [15] also found a significant difference between the children and parents on the "Oral symptoms" (39.10 vs.40.52), "School" (14.64 vs. 14.06), and on the "Emotional well-being" (31.22 vs. 29.31) sub-scale. So, the parents’ expectations about their children’s OHRQoL differed from their children’s reports.

As indicated by Nolte et al. [16], the parent-child test scores were high and showed great OHRQoL. Caregivers scored substantially higher than their kids on the "Oral Symptoms" (39.28 vs. 36.69) sub-scale and fundamentally lower on the "Functional Well-being" (24.64 vs. 25.32) sub-scale than their kids.

While both the studies by Ward et al. [17] done in the USA and Abebe et al. [18] done on

Ethiopian children/parents, reported that no statistically significant discrepancies between the responses of orofacial cleft children and their caregivers for OHRQoL as a whole for both studies or any of the sub-scales for the research performed by Ward et al. [17]. In contrast, there were some significant differences found in the study conducted by Abebe et al. [18] on the sub-scale levels. The correlation coefficient for the "Emotional well-being" was found to be high, followed by "oral symptoms" and "functional well-being" sub-scales. While the correlation in the "school environment" was found to be relatively low. The findings in this study showed good OHRQoL, which was demonstrated by the high overall score parents and patients obtained.

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Table 5. Overall and sub-scale COHIP scores and differences between children and parents (mean ± SD)

COHIP - Child Oral Health Impact Profile ; SD - standard deviation

Subscale

Study

Oral Health Functional

well-being

Social-Emotional well-being

School Environment

Self -Image Peer interaction Overall

COHIP Score

Child Parents Child Parents Child Parents Child Parents Child Parents Child Parents Child Parents

Konan et al. [14] 35.89 ±6.10 36.39 ±7.08 23.59 ±4.37 23.25 ±4.91 31.25 ±6.13 31.12 ±7.21 17.29 ±2.69 17.64 ±2.98 19.75 ±5.50 21.19 ±5.58 - - 127.8 ±17.16 130.06 ±20.55 Bos et al.[15] 39.10 ± 4.48 40.52 ± 5.04 24.98 ± 3.82 24.51 ±4.36 31.22 ±5.21 29.31 ±7.02 14.64 ±0.90 14.06 ±2.08 - - 9.26 ± 1.09 8.97 ±1.7 119.29 ±11.55 117.67 ±15.36 Nolte et al. [16] 36.69 ± 6.16 39.28 ± 5.92 25.32 ±3.75 24.64 ±4.2 30.87 ±5.17 30.27 ±6.01 18.67 ±1.63 18.63 ±1.76 - - 9.32 ±1.22 9.10 ±1.26 120.53 ±13.14 121.75 ±14.47 Ward et al. [17] 24.8 ±5.9 23.90 ±6.2 17.70 ±4.7 16.10 ±4.6 24.30 ±6.7 21.50 ±7.8 13.1 ±3 13.2 ±2.7 15.7 ±4.3 15.5 ±4.3 - - 95.6 ±18.3 90.2 ±19 Abebe et al. [18] 39.14 ±5.81 39.90 ±7.47 35.60 ±6.23 33.58 ±6.95 39.88 ±9.22 40.00 ±10.11 16.93 ±4.61 16.61 ±4.37 - - 24.60 ±5.88 26.39 ±5.29 155.56 ±26.2 155.51 ±30.79 Agnew et al. [11] - - - -Eslami et al. [19] - - - -Broder et al. [20] - - -

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3.3 Evaluation of the differences between different age groups according to the COHIP index

Overall and sub-scales COHIP scores comparisons by age groups are presented in Table 6.

The group of children and young adolescents had the same overall COHIP and all sub-scale ratings, according to Konan et al. [14]. The explanation, there was no difference in OHRQoL between children and young adolescents in this study, could be attributed to the small age range of the two sample groups.

While according to Bos et al. [15], differences between patients younger and older than 12 years old were found. Cleft patients aged 12 and older scored significantly lower on the sub-scales of " Emotional health" (30.14 vs. 32.36) and "Oral symptoms" (38.04 vs. 40.04). Their presented results appear to show that younger patients are less conscious of themselves and their symptoms than olderpatients. Cleft patients aged 12 and older have more experience of treatment, and this may also explain the difference in age they found.

Nolte et al. [16] also made a comparison between different age groups, but no values were mentioned in their article.

Nolte et al. and Ward et al. [16-17] did not find any significant differences between the different age groups for the COHIP overall score or any of its sub-scales.

While Eslami et al. [19] found that there were no significant differences between patients younger than and over 12 years in the overall COHIP and its sub-scale ratings. This indicates younger patients were conscious of themselves and their symptoms.

According to Agnew et al. [11], he found that the older patients had lower overall and sub-scale scores than the younger ones.

Broder et al.[20] did an age classification different from the usual one that was used for the instrument. They split them up into three age groups, from 7 to 9, from 10 to 13, and 14-19 years. Their study found no significant differences in OHRQoL between cleft patients in different age groups. Orofacial clefts had a more substantial impact on "social-emotional well-being" in 14-19-year-olds (23.2) than in 7-9-year-olds (26.3) or 10-13-year-olds (25.5).

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Table 6. Overall and sub-scales COHIP scores comparisons by age groups (mean ± SD)

Study Age groups Oral Health Functional Well- being

Social/Emotional Well-being

School Environment

Self -Image Peer interaction Overall COHIP Score Konan et al. [14] Child

( 8-12) 35.86 ± 6.00 23.70 ± 4.73 31.58 ± 6.72 17.32 ± 2.81 19.52 ± 6.13 - 127.99 ± 18.6 Adolescent (12-15) 35.93 ± 6.23 23.51 ± 4.06 30.97 ± 5.61 17.27 ± 2.6 19.95 ± 4.92 - 127.63 ± 15.94

Bos et al. [15] Child ( 8-12) 40.04 ± 4.57 25.16 ± 3.54 32.36 ± 4.65 14.69 ± 0.62 - 9.36 ± 0.93 121.77 ± 10.18 Adolescent (12-15) 38.04 ± 4.34 24.78 ± 4.12 30.14 ± 5.47 14.60 ± 1.22 - 9.14 ± 1.25 116.19 ± 12.36

Ward et al. [17] Child ( 8-10) 25.5 ± 6 15.9 ± 5.1 24.0 ± 7.8 12.5 ± 4.1 15.6 ± 4.4 - 93.3 ± 20.3 Adolescent (11-14) 25.2 ± 5.9 18.7 ± 3.8 25.3 ± 5.5 13.6 ± 1.6 15.8 ± 4.3 - 98.6 ± 15.1 Eslami et [19] Child ( 8-12) 22.80 ± 4.6 20.93 ± 5.89 24.80 ± 9.59 6.73 ± 4.38 - 12.00 ± 7.63 87.27 ± 23.49 Adolescent (12-15) 24.86 ± 7.32 21.77 ± 7.54 29.97 ± 11.16 7.06 ± 3.95 - 12.80 ± 6.23 96.46 ± 28.92

Agnew et al. [11] Child ( 7-10) 12.8 11.8 29.0 - - - 53.6 Adolescent (11-14) 11.7 10.07 26.9 - - - 49.2 Nolte et al. [16] - - - - Abebe et al. [18] - - - - Broder et al. [20] - - - -

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23

3.4 Evaluation of the differences between different genders according to the COHIP index

Overall and sub-scales COHIP scores comparisons by gender are presented in Table 7.

According to Konan et al. [14], in every sub-scale and the overall COHIP, girls had slightly better scores and more positive OHRQoL than boys. Despite the general accord of the relationship between females, especially those with oral clefts, and affectability about the variables identified with HRQoL, there was no solid proof in this study.

In the studies performed by Bos et al. in 2011 [15], Agnew et al. [11] in 2017, and Broder et al. [20] in 2012, they found that there was no impact of gender on the COHIP scores.

Nolte et al. [16] found that females, on the "Functional Well-being" (26.11 vs. 24.68) and "School" sub-scales (18.97 vs. 18.43) show significantly higher OHRQoL than males. The gender differences that were found in this study were different from those found in previous studies (Bos and Prahl) [15].

Eslami et al.[19] found a significant difference between girls and boys with the cleft lip and palate on the sub-scale "Emotional well-being" (23.35 vs. 25.21). It means that girls’ quality of life has been affected more by oral health. However, a significant sex difference in the overall COHIP was not found in their study on Iranian patients. This result is in agreement with Bos and Prahl’s (2011) [15] study on OHRQoL in Dutch children with CLP.

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24

Table 7. Overall and sub-scales COHIP scores comparisons by gender (mean ± SD)

COHIP - Child Oral Health Impact Profile ; SD - standard deviation

Subscale

Study

Oral Health Functional well-being Social-Emotional well-being

School Environment

Self -Image Peer interaction Overall

COHIP Score

Females Males Females Males Females Males Females Males Females Males Females Males Females Males

Konan et al. [14] 36.13 ± 5.91 35.66 ±6.32 24.10 ± 4.08 23.09 ±4.6 31.42 ± 6.4 31.10 ± 5.91 17.42 ± 2.7 17.16 ±2.7 20.39 ±5.51 19.10 ±5.45 - - 129.47 ±16.52 126.11 ±17.74 Bos et al. [15] 38.22 ± 4.82 39.69 ± 4.31 25.06 ± 3.41 24.91 ±4.14 30.69 ± 5.35 31.74 ±5.01 14.54 ± 1.11 14.72 ±0.68 - - 9.23 ±1.23 9.28 ±0.97 118.20 ±11.63 119.81 ±11.54 Nolte et al. [16] 36.66 ±5.76 36.73 ±6.49 26.11 ±3.75 24.68 ±3.73 31.21 ±5.35 30.60 ±5.04 18.97 ±1.61 18.43 ±1.62 - - 9.20 ±1.47 9.42 ±0.96 121.79 ±13.39 119.54 ±12.94 Agnew et al. [11] 12.2 12.4 11.5 11.3 26.6 27.4 - - - 50.4 51.1 Eslami et al. [19] 23.35 ±7.32 25.21 ±5.83 22.38 ±8.16 20.58 ± 5.6 31.65 ±10.78 24.92 ±10.07 7.96± 4.56 5.88± 3.13 - - 13.27 ±6.87 11.79 ±6.36 98.62 ±30.06 88.38 ± 23.91 Broder et al. [20] 25.5 25.1 18.2 17.8 24.4 25.4 13.3 13.4 17.1 16.4 - - 99.2 98.6 Ward et al. [17] - - - -Abebe et al. [18] - - -

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-25

3.5 Evaluation of the differences between different cleft types according to the COHIP index

Comparison of COHIP scores among cleft types are presented in Table 8

According to Konan et al. [14], cleft lip patients with (out) alveolus CL(A) had more positive overall domains of COHIP and "functional well-being" than patients with unilateral (UCLP) and bilateral cleft lip/palate (BCLP). The type of cleft, however, did not have a significant effect on the oral health-related quality of life of patients in general. However, this study did not discover any considerable differences in OHRQoL among the cleft forms.

Bos et al. [15] showed a significant difference between different types of clefts and the sub-scale "functional well-being", for which the CL(A) category had the highest scores.

Nolte et al. [16] studied the various cleft forms, and children with a BCLP had lower sub-scale scores on "Functional well-being" and "School", but no values where included in their study. For Eslami et al. [19], they found that the BCLP category included more severe cases, and found no significant difference between the two forms of clefts in the overall COHIP and its sub-scales scores.

Those with BCLP reported worse "Functional well-being" scores than anyone affected by other types of clefts, according to Agnew’s et al. [11] research.

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26

Table 8. Comparison of COHIP scores among cleft types (mean ± SD)

Scale

Study

Oral Health Functional Well- being Social/Emotional Well- being

School Environment Self -Image Peer interaction Overall COHIP Score CL (A) CP UCL BCLP CL (A) CP UCL BCLP CL (A) CP UCL BCLP CL (A) CP UCL BCLP CL (A) CP UCLP BCLP CL (A) CP UCLP BCLP CL (A) CP UCLP BCLP Konan et al.[14] 37.56 ± 4.7 - 35.10 ± 6.43 36.44 ± 6.14 25.92 ± 3.57 - 23.17 ± 4.43 22.29 ± 4.32 33.09 ± 5.52 - 30.99 ± 6.22 29.68 ± 6.18 18.07 ± 2.07 - 17.09 ± 2.85 17.07 ± 2.73 20.9 6 ± 5.96 - 19.53 ± 5.28 18.68 ± 5.38 - - - - 135.59 ± 15.06 - 125.87 ± 17.42 124.16 ± 16.08 Bos et al. [15] 40.44 ± 4.35 37.9 ± 4.92 38.88 ± 4.14 38.94 ± 4.61 26.46 ± 3.31 23.85 ± 4.62 24.52 ± 3.13 24.78 ± 3.59 30.74 ± 5.59 30.91 ± 4.57 32.06 ± 5.23 31.78 ± 5.34 14.74 ± 0.76 14.48 ± 1.28 14.66 ± 0.7 14.72 ± 0.57 - - - - 9.34 ± 1.02 9.47 ± 0.98 9.09 ± 1.2 9.00 ± 1.2 121.65 ± 11.3 116.29 ± 11.61 119.2 ± 11.38 119.25 ± 12.11 Eslami et al. [19] - - 23.88 ± 5.9 24.63 ± 7.48 - - 23.31 ± 5.69 19.58 ± 7.92 - - 31.15 ± 11.46 25.46 ± 9.58 - - 7.23 ± 4.1 6.67 ± 4.05 - - - 13.42 ± 7.38 11.63 ± 5.67 - - 99 ± 27.85 87.96 ± 26.49 Agnew et al. [11] 14.1 12 12.3 11.6 13.1 10.8 12 10.6 27.2 29.7 26.2 25.2 - - - 54.5 52.5 50.5 47.4

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4. DISCUSSION

This systematic review integrated proof about OHRQoL ideas from 8 studies of patients with CL/P using the COHIP questionnaires that both the parents and their children replied to. This study uses a more comprehensive approach, including capturing the viewpoint of patients using questionnaires to assess their quality of living. This review focused specifically on assessing the quality of life of CL/P patients concerning various aspects of their lives from the viewpoint of the patient and/or the patient's parent.

According to a 2011 study by Klassen et al. they proclaimed that as of recently, there were no patient-reported outcome (PRO) instruments intended to address the problems and worries of CL/P patients. However, an assortment of these instruments has been altered for use in children with different dental, oral and craniofacial conditions, where COHIP is one of these instruments [21-22].

While addressing OHRQoL, it is important to consider the caregiver(s), because although a reliable approach can be used to determine the OHRQoL of the child, it is the caregiver(s) that needs to understand how to avoid health problems, how to teach the child about the reality of their problems and how to decide whether there is a problem that needs intervention. In general, parents have a small to moderate cooperation with the QoL scores of their infant (Achenbach et al., 1987 [23]; Eiser and Morse, 2001 [24]). Parents and their interaction with the OHRQoL of their child often differed according to different publications. Abebe et al.[18] and Ward et al. [17] noted that there was a high degree of overlap between the ratings of both parents and children, and they concluded that there was no significant difference in all sub-scales and total COHIP scores between the children and their parents about their OHRQoL. While the COHIP surveys of children and parents indicate strong concordance in all the publications of the studies, there were major variations in the "Oral symptoms" and "Functional well-being" sub-scales. Patients ranked below their parents on the "Oral Health" sub-scale and higher on the "Functional well-being" sub-scale. It suggests that parents underestimate the oral health of their children and the signs they feel, example, pain or discomfort of their teeth, or breathing problems, or even the appearance or color of their teeth or the gaps between them. The parents often overestimate the practical inconveniences of possessing clefts such as having trouble in chewing, sleeping, cleaning, and talking. Wilson-Genderson et al. (2007) [8] found that their study of craniofacial, orthodontic, and pediatric children had small to medium levels of caregiver agreements. They observed that craniofacial participants appeared to score their OHRQoL higher than the responses of their caregivers. In contrast, pediatric and orthodontic

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28 patients were more likely to agree with the responses of their caregivers, or lower their OHRQoL. Many people with orofacial clefts have views on their looks, which give their quality of life an important aspect.

Only a small number of studies of children and adolescents with clefts have looked into age-related variations in OHRQoL. Although the study by Broder and Wilson-Genderson [8] observed age-related differences when mixing participants from the craniofacial, orthodontic, pediatric, and control groups, discrepancies were not identified only within the craniofacial community. Data from a study conducted by Damiano et al. [6] revealed age-related variations in HRQoL in orofacial cleft preadolescent infants. Their study results show that the variation in external appearance associated with lip activity is of greater relative importance as the infant gets closer to puberty. Older kids, however, will have more understanding in therapy than younger patients, though, and this may also explain the difference in age they observed. According to Damiano [6], the relationship between the cleft type and quality of life was more nuanced, but it differed according to a child's age group. The HRQoL scores among children with CL or CL/P increased from ages 2 to 4 to ages 5 to 7. However, they deteriorated for children aged 8 to 12, a time when the physical appearance becomes more significant as regards social interaction and self-assessment. Increasing ratings for children with CP may be correlated with expression changes that could occur following surgery and speech therapy. According to the studies conducted by Konan et al.[14], Eslami et al.[19] and Nolte et al. [16] observed that, in any of the sub-scales or total COHIP ratings, there were no significant age gaps between children under 12 and older than 12. It suggests that younger patients were also mindful of their symptoms and themselves. Ward et al. [17] reported comparable findings. Still, the main discrepancy was that he had his sample population aged 8-18. The clefts had a stronger effect on the 15-18-year-old on the "Social-Emotional Well-being" score, likely because fears about the presence and views of others are becoming more important as children with orofacial clefts reach the age of sexual relationships. In cleft patients aged 14-19 years, Broder et al. [20] have reported poorer "emotional well-being" than younger cleft patients in the USA. Both research by Bos et al.[15] and Agnew et al. [11] showed that the older patients scored lower on all sub-scales, and COHIP overall, specifically the "emotional well-being" result. From these findings, one can infer that there may be no significant differences between different age groups. This is because the children are more conscious of themselves and their signs and symptoms, but not the case when taking "emotional well-being" and "oral health" sub-scales into effect. This could be because the children are less conscious of their symptoms and themselves when they are still young.

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29 Gender impacts a child's biological, personal, and social growth within a community and culture. Gender-to-HRQoL partnerships typically occur. The literature includes many studies on gender effects on OHRQoL. There were no substantial gender differences between males and females on either sub-scale or total COHIP scores in the analysis conducted by Broder et al. in 2012 [20]. We can find similar results in the experiments conducted by Bos et al. in 2011 [15] and by Agnew et al. in 2017 [11]. However, that was not the case for the other studies being reviewed in this analysis where females in almost every sub-scale and average COHIP scores had a better score than males. Nevertheless, the sub-scale of "Oral Health" shows that males had higher scores suggesting that females usually have greater OHRQoL than males except for oral health status. Similar to these findings, McGrath and Bedi [25] have stated that women were more insecure and self-conscious, and also felt more discomfort and unhappiness relative to men because of their oral health. The "emotional well-being" sub-scale showed the most striking variations out of all the other sub-scales where females had higher scores in the Konan et al.[14], Nolte et al. [16] and Eslami et al. [19] studies. Though males had better scores in the Bos et al. [15], Agnew et al.[11], and Broder et al.[20] studies. A work conducted by Broder and colleagues found that females had reduced socio-emotional well-being relative to males with increasing age. Perhaps the findings of this research may help understand why the "emotional well-being" sub-scale has a gender difference.

When comparing patients with different types of clefts, we can infer that patients with cleft lips with or without alveolus CL(A) have better OHRQoL as compared with other types of clefts. Bilateral CL/P had the lowest scores in the sub-scales, especially on the sub-scale of "Functional Well-Being. " This can be explained because BCLP causes a severe burden on the life of the child and the fact that these children are likely to have more functional restrictions and aesthetic complaints [26]. So, this finding can be explained as to why the BCLP had the lowest score out of all types of cleft because it causes great difficulties for the patients while chewing, teeth brushing, and speech articulation.

This systematic review has had limitations, and they should be listed. The COHIP instrument was built to research the children’s OHRQoL between the ages of 8 and 15 years. Some of the reported trials, however, went beyond this context and included older subjects, so this could have provided many different results when comparing the participants ' ages and presenting their relationship to the OHRQoL. Gender could also have been categorized according to age groups so that the results obtained in the analysis will be understandable. Another drawback is that these experiments have different types of cleft classifications, which might also hamper the results. Nearly all of the researches included in this systematic review were cross-sectional

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30 analysis, so this could have led to insufficient information during the outcome evaluation and bias risk assessment. In future examinations, risk factors for a diminished QoL ought to be recognized, and ways to deal with updated flexibility for adolescents with a CL/P ought to be explored [27-28].

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31

5. CONCLUSIONS

1. The children’s OHRQoL varies from those of their parents, particularly on the oral and functional well-being sub-scales.

2. Younger patients had better scores of OHRQoL on the emotional well-being and oral health sub-scales, mainly.

3. Females had a better OHRQoL than males except for oral health.

4. The quality of life of children varies between various types of clefts where out of all cleft forms, the bilateral cleft lip and palate patients has been the most affected.

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6. PRACTICAL RECOMMENDATIONS

Through the discoveries of this orderly examination, we may presume that the COHIP instrument is fitting for children with CL/P to test the OHRQoL. As per this deliberate audit, it is smarter to utilize the COHIP test in longitudinal investigations that would be a need for imminent prospective analysis. Since there were a few resemblances between the responses of the patients and their caregivers, the utilization of indirect parental information for extra data when child information about their OHRQoL is missing, can be useful. Perhaps utilizing a shorter adaptation of the COHIP survey may help recognize patients with lower OHRQoL scores and assist them with accomplishing the necessary measure of psychological advising.

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7. REFERENCES

1. Mossey PA, Modell B. Epidemiology of Oral Clefts 2012: An International Perspective. Front Oral Biol. 2012;16:1-18.

2. Peterson LJ, Ellis E, Hupp JR, Tucker MR. Contemporary oral and maxillofacial surgery. 3rd ed. 1998:656–79.

3. Gorlin R J, Cohen M Jr, Hennekam RCM. Syndromes of the head and neck. Oxford University Press, New York. 2001

4. Homer CJ, Kleinman LC, Goldman DA. Improving the quality of care for children in health systems. Health Serv Res. 1998;33(4 Pt 2):1091-109.

5. Topolski T, Edwards T, Patrick D. Quality of Life: How Do Adolescents with Facial Differences Compare with Other Adolescents? Cleft Palate Craniofac J. 2005;42(1):25-32. 6. Damiano P, Tyler M, Romitti P, Momany E, Jones M, Canady J et al. Health-Related Quality of Life Among Preadolescent Children with Oral Clefts: The Mother's Perspective. Pediatrics. 2007;120(2):e283-90.

7. Raposo-do-Amaral C, Kuczynski E, Alonso N. Qualidade de vida de crianças com fissura labiopalatina: análise crítica dos instrumentos de mensuração. Revista Brasileira de Cirurgia Plástica. 2011;26(4):639-644.

8. Broder H, Wilson-Genderson M. Reliability and convergent and discriminant validity of the Child Oral Health Impact Profile (COHIP Child’s version). Community Dent Oral Epidemiol. 2007;35s(1):20-31.

9. Broder H, Wilson-Genderson M, Sischo L. Examination of a Theoretical Model for Oral Health–Related Quality of Life Among Youths with Cleft. Am J Public Health. 2014;104(5):865-871.

10. Shrive F., Stuart H, Quan H, Ghali W. Dealing with missing data in a multi-question depression scale: a comparison of imputation methods. BMC Med Res Methodol. 2006;6(1) 11. Agnew C, Foster Page L, Hibbert S. Validity and reliability of the COHIP-SF in Australian children with orofacial cleft. Int J of Paediatr Dent. 2017;27(6):574-582.

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34 12. Moher D, Liberati A, Tetzlaff J, Altman DG, PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: The PRISMA statement. PLoS Med 2009;6:(7):e1000097

13. Downes MJ, Brennan ML, Williams HC, Dean RS. Development of a critical appraisal tool to assess the quality of cross-sectional studies (AXIS). BMJ Open. 2016;6(12): e011458. 14. Konan P, Manosudprasit M, Pisek P, Pisek A, Wangsrimongkol T. Oral Health-Related Quality of Life in Children and Young Adolescent Orthodontic Cleft Patients. J Med Assoc Thai. 2015;98(s7):S84-91.

15. Bos A, Prahl C. Oral health-related quality of life in Dutch children with cleft lip and/or palate. Angle Orthod. 2011;81(5):865-871.

16. Nolte FM, Bos A, Prahl C. Quality of Life Among Dutch Children with a Cleft Lip and/or Cleft Palate: A Follow-Up Study. Cleft Palate Craniofac J. 2019;56(8):1065-1071.

17. Ward JA, Vig KW, Firestone AR, Mercado A, da Fonseca M, Johnston W. Oral Health– Related Quality of Life in Children with Orofacial Clefts. Cleft Palate Craniofac J. 2013;50(2):174-81.

18. Abebe ME, Deressa W, Oladugba V, Owais A, Hailu T, Abate F et al. Oral Health–Related Quality of Life of Children Born with Orofacial Clefts in Ethiopia and Their Parents. Cleft Palate Craniofac J. 2018;55(8):1153-1157.

19. Eslami N, Majidi MR, Aliakbarian M, Hasanzadeh N. Oral Health-Related Quality of Life in Children with Cleft Lip and Palate. J Craniofac Surg. 2013;24(4):e340-3.

20. Broder H, Wilson-Genderson M, Sischo L. Health Disparities Among Children with Cleft. Am J Public Health. 2012;102(5):828-830.

21. Pusic AL, Lemaine V, Klassen AF, Scott AM, Cano SJ. Patient-Reported Outcome Measures in Plastic Surgery: Use and Interpretation in Evidence-Based Medicine. Plast Reconstr Surg. 2011;127(3):1361-7.

22. Klassen A, Tsangaris E, Forrest C, Wong K, Pusic A, Cano S et al. Quality of life of children treated for cleft lip and/or palate: A systematic review. J Plast Reconstr Aesthet Surg. 2012;65(5):547-57.

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35 23. Achenbach TM, McConaughy SH, Howell CT. Child/adolescent behavioral and emotional problems: Implications of cross-informant correlations for situational specificity. Psychol Bull. 1987;101(2):213–32.

24. Eiser C, Morse R. Can parents rate their child's health-related quality of life? Results of a systematic review. Qual Life Res. 2001;10(4):347-57.

25. McGrath C, Bedi R. Gender variations in the social impact of oral health. J Ir Dent Assoc 2000;46(3):87-91

26. Broder HL, Wilson-Genderson M, Sischo L. Oral health-related quality of life in youth receiving cleft- related surgery: self-report and proxy ratings. Qual Life Res. 2016;26(4):859-867.

27. Feragen KB, Kvalem IL, Rumsey N, Borge AI. Adolescents with and without a facial difference: The role of friendships and social acceptance in perceptions of appearance and emotional resilience. Body Image. 2010;7(4):271-9.

28. Broder H. Using Psychological Assessment and Therapeutic Strategies to Enhance Well-Being. Cleft Palate Craniofac J. 2001;38(3):248-254.

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36

8.ANNEXES

8.1.Annex 1

Quality of life among children with cleft lips and palates: a critical review of measurement instruments.

World Health Organization Quality of Life assessment instrument (WHOQOL), Pediatric Quality of Life Inventory (PedsQL), 36-Item Short Form Health Survey Questionnaire (SF-36), Rand-36, Oral Health Impact Profile (OHIP), Child Oral Health Impact Profile (COHIP), Michigan Oral-Health Related Quality of Life scale (MOHRQoL), KINDL, Child Perceptions Questionnaire (CPQ), Children’s Health and Illness Profile– Adolescent Edition (CHIP-AE); Child Health Questionnaire Parent Form 28 (CHQ-PF28). Youth Quality of Life Instrument – Craniofacial Surgery (YQOL-CS), which means to survey, from a personal point of view, the effect of the surgery on an individual’s life; and the Youth Quality of Life Instrument – Facial Differences (YQOL-FD). The impact on family-scale questionnaire, The Cleft Research Questionnaire (CRQ), and the VELO questionnaire.

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8.2. Annex 2

The Child Oral Health Impact Profile (COHIP) questionnaire

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38

8.3. Annex 3

Appraisal tool for Cross-Sectional Studies (AXIS)

Critical Appraisal (CA) is utilized to survey investigated papers and to pass judgment on the unwavering quality of the examination being exhibited in the article. CA additionally helps in studying the value and significance of the review. There are many critical territories to CA, including evaluating the reasonableness of the investigation to respond to the theorized question and the chance of bringing bias into the examination. Recognizing these critical territories in CA requires excellent revealing of the study. If the test is inadequately announced, the appraisal of reasonableness and bias gets troublesome.

The accompanying evaluation instrument was created for use in assessing observational cross-sectional examinations. It is intended to address issues that are regularly clear in cross-cross-sectional investigations and to help the pursuer while surveying the quality of the study that they are appraising. The tool aims to help orderly understanding of a cross-sectional examination and to illuminate choices about the quality regarding the investigation being tested.

The evaluation instrument accompanies a graphic assistance content which gives some foundation information and clarification concerning what the inquiries are posing. The interpretations are intended to advise why the investigations are significant.

The examination instrument has zones to record a "yes," "no," or "don't know" answer for each question, and there is space for short remarks also.

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