The Cleft Palate–Craniofacial Journal 53(2) pp. 187–196 March 2016 Ó Copyright 2016 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Evaluation of Facial Appearance in Patients With Cleft Lip and Palate by Laypeople and Professionals: A Systematic Literature Review Shiwen Zhu, B.D.S., M.D.S., Jayakumar Jayaraman, B.D.S., Ph.D., M.Paed. R.C.S. (Edin), Balvinder Khambay, F.D.S. R.C.S. (Eng), M.Orth. R.C.S. (Edin), Ph.D. Objective: To identify whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate (CLP) similarly based on viewing full facial images. Design: Several electronic databases were searched. A hand search was performed among reference lists and relevant journals. Studies that assessed the full facial appearance of patients with CLP based on two-dimensional (2D) photographs, 3D images, or clinical examination by laypeople and professionals using a visual analog scale (VAS) or a categorical rating scale were included. Two authors independently assessed articles using methodologic-quality scoring protocol. Results: Eleven articles were included in qualitative synthesis, including four high-level and seven moderate-level papers. Three studies found that laypeople were more critical than professionals, three found there was no significant difference between laypeople and professionals, and five reported that professionals were more critical than laypeople when assessing facial appearance of patients with CLP. Conclusions: It still remains unknown whether laypeople are more or less critical than professionals when rating facial appearance of patients with repaired CLP. Professionals are more familiar with the esthetic outcomes and difficulties of treating patients. The opposite maybe true for laypeople; this disparity between what is achievable by professionals and what is expected by laypeople may be a source of dissatisfaction in facial appearance outcome. Further well-designed studies should be carried out to address this question and the clinical significance of the difference in rating scores for patients with CLP. KEY WORDS:
cleft lip and palate, facial appearance, laypeople, professionals, systematic review
The first of these is a primary lip repair, which is carried out to improve function, speech, and esthetics. Inadequate correction of facial esthetics, in particular nasolabial symmetry is thought to be a source of emotional distress and dissatisfaction among this group of patients (Gkantidis et al., 2013). This may be the reason for further surgical revision procedures (Foo et al., 2013). In the present era of patient-centered interdisciplinary management, in addition to the patient, professionals and laypeople may also indirectly affect the decision for further surgery. Even though the patient’s opinion is paramount in requesting further surgery to improve his or her perceived quality of life (Chung et al., 2013), the opinion and support of laypeople such as friends and peers have been shown to have a positive effect on the psychological well-being of the individual (Moss, 1997; Pedersen et al., 2007). Facial attractiveness is of critical importance in various social interactions (Little et al., 2007; Coetzee et al., 2014). Social interactions of patients primarily depend on perceptions of the wider community, which mainly consists of laypeople or peers (Williamson, 1999; Rankin and Borah, 2003). It has been shown that patients with cleft lip and palate often experience negative social interactions as a result of an unattractive facial appearance (Chung et al.,
Cleft lip and palate is the most common congenital dysmorphology affecting the oral and maxillofacial region (Stec et al., 2007). The prevalence of cleft lip and palate varies between countries, socioeconomic status, and ethnicities (Shaw, 2004). The average worldwide prevalence of patients with cleft lip with or without cleft palate has been reported as 7.94 per 10,000 births (Tanaka et al., 2012). Among the ethnicities, the Chinese have the highest prevalence of cleft lip and palate, affecting 16.63 per 10,000 births during 1996 to 2005, with an upward trend over time (Dai et al., 2010). In order to address the facial dysmorphology of patients with cleft lip and palate, a series of surgical interventions are undertaken at specific times during the child’s development.
S. Zhu and Dr. Jayaraman are research postgraduate students and Dr. Khambay is Clinical Associate Professor, Discipline of Orthodontics and Paediatric Dentistry, Faculty of Dentistry, The University of Hong Kong, Hong Kong, Hong Kong SAR. Submitted June 2014; Revised September 2014, November 2014; Accepted December 2014. Address correspondence to: Dr. Balvinder Khambay, 2A, Prince Philip Dental Hospital, 34 Hospital Road, Hong Kong. E-mail [email protected]. DOI: 10.1597/14-177 187
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2013). Individuals with congenital craniofacial conditions often experience discrimination in an employment and social setting related to lower satisfaction with facial appearance, quality of life, and self-esteem (Sarwer et al., 1999). This highlights the need to involve laypeople as potential peers in assessing facial appearance outcome. Numerous studies have investigated the outcome of postsurgical facial appearance in patients with cleft lip and palate based on the views of laypeople and professionals. Some studies have focused on cropped photographs of the isolated nasolabial region (Bongaarts et al., 2008; Kim et al., 2009, 2011; Mani et al., 2010), while others evaluated the full frontal facial image of patients with cleft lip and palate (Lo et al., 2002; Papamanou et al., 2012; Chung et al., 2013; Gkantidis et al., 2013; Offert et al., 2013). The use of cropped photographs may not be appropriate as they do not indicate total facial harmony and may therefore be misleading. The esthetic outcome of the repaired cleft lip should not be viewed in isolation but should be based on overall facial appearance as it is in other facial dysmorphologies (i.e., orthognathic patients). There is agreement that laypeople and professionals rate the facial appearance of patients with cleft lip and palate consistently lower than noncleft individuals (Eichenberger et al., 2014). However, when assessing the facial appearance of individuals with clefts there are conflicting opinions between laypeople and professionals. Some studies have reported professionals are more critical (Eliason et al., 1991; Chung et al., 2013; Foo et al., 2013; Offert et al., 2013), while others reported that laypeople were more critical (Papamanou et al., 2012; Gkantidis et al., 2013; Eichenberger et al., 2014). The aim of this systematic review was, therefore, to determine whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial images. METHODS Eligibility Criteria The selected studies compared full facial appearance of cleft repair as judged by laypeople and professionals. Studies that met the following criteria were included: (1) patients with cleft lip with or without cleft palate; (2) outcome of facial appearance assessed by laypeople and professionals; (3) outcome measure based on a visual analog scale (VAS) or a categorical rating scale; (4) use of two-dimensional (2D) photographs, 3D images, or real patients; and (5) assessment of full facial images. Exclusion criteria were as follows: (1) patients without cleft lip; (2) only one group of assessors; (3) use of video images; (4) reviews, letter to editors, expert opinions, and case reports; and (5) partial facial images, i.e., cropped or isolated surgical cleft region.
Information Resources PubMed (all available articles until week 2 of June 2014), The Cochrane Library (1999 to week 2 of June 2014), Web of Science (1956 to week 2 of June 2014), Scopus (1999 to week 2 of June 2014), and EMBASE (1980 to week 2 of June 2014) were searched to identify relevant articles. The reference lists in these relevant articles were retrieved manually. The articles in Cleft Palate-Craniofacial Journal (from January 2004 to May 2014) and American Journal of Orthodontics and Dentofacial Orthopedics (from January 2004 to June 2014) were also manually searched based on their relevance to facial appearance and the patients with cleft lip and palate. No language limit was set. Review Question The patients, intervention, comparator, and outcome (PICO) structure was applied to form the research question. In this systematic review, patients are those with cleft lip and palate. The comparison will be carried out between the professionals and laypeople. The outcome will be the difference in evaluation of facial appearance by professionals and laypeople. Therefore, the review question is whether professionals and laypeople rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial images. Search Strategy Search terms were identified to fit the research question, including cleft, laypeople, professionals, assess, face, and appearance. Based on these search terms, the search strategy was as shown in Table 1. PRISMA guidelines were used to conduct the present systematic review (Liberati et al., 2009). Initially, two authors (S.Z. and B.K.) independently searched the databases on the basis of title and abstract. Papers that met the eligibility criteria were included. If the eligibility of papers could not be identified by title and abstract, the full-text articles were read by the reviewers. An additional search was performed manually utilizing the reference lists of included papers and relevant journals for identifying eligible articles. Quality Assessment To evaluate the methodologic quality of each article, an eight criteria-based scoring protocol was used, modified from Lagravere et al. (2005), as shown in Table 2. Two authors independently completed the information extraction sheet for the included papers, which was designed on the basis of the review question and PICO structure, as shown in Table 3. Disagreement between
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TABLE 1
Search Strategy for Electronic Databases
PubMed ((((((cleft) OR harelip)AND (((((laypeople) OR layperson*) OR layrater*) OR non-profession*) OR general public) OR lay public) AND (((((profession*) OR clinician*) OR orthodonti*) OR surgeon*) OR maxillofacial surgeon*) OR oral surgeon* ) AND ((((((assess*) OR evaluat*) OR prefer*) OR perceive) OR perception) OR view) OR psycholog* ) AND (((((face) OR facial) OR dentofacial) OR profile) OR lateral) OR cephalometric*) AND ((((attract*) OR aesthetic*) OR esthetic*) OR appearance) OR beauty Cochrane Library (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) Web of Science (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) Scopus (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) EMBASE (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty)
two authors was resolved by discussion and consensus. When two authors could not reach an agreement, a third reviewer (J.J.) was asked and consensus reached. According to the total score, studies were classified into low (score ,6), moderate (score .6 and ,10), and high (score .10) level of quality, as shown in Table 4 (Lagravere et al., 2005; Baratieri et al., 2011). RESULTS In total, 112 studies were identified following the database searches. Eleven articles were finally included in qualitative synthesis. The retrieval of PubMed, The Cochrane Library, Web of Science, Scopus, and EMBASE provided 19, 5, 18, 58, and 12 records, respectively. After removing duplicates and ineligible articles, 11 publications
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TABLE 2 Methodologic-Quality Scoring Protocol (Maximum Score, 12 Points) Based on Lagravere et al. (2005) I. Study design (8) A. Full face photograph (2D or 3D), including frontal side (1) B. Cleft patients: homogeneous (2) or heterogeneous (1) C. Viewed by professionals (2) D. Viewed by laypeople: nonmedical background (2) or medical background (1) E. Viewed on two separate occasions (1) II. Study measurements (4) A. Rating system: VAS scale 0 to 100 mm (2) or categorical scale (3-, 4-, 5-, 6-, 9-, and 10-point scale) (1) B. Intrapanel agreement (1) C. Interpanel agreement (1)
remained. Seven additional records were identified through reference linkage by hand searching. After reading the fulltext articles, 11 of them were finally included in qualitative synthesis. The PRISMA flow diagram is shown in Figure 1. Number and Type of Patients With Clefts The majority of studies evaluated the facial appearance of unilateral cleft lip and palate; only one study evaluated patients with bilateral cleft lip and palate (Lo et al., 2002). However, three of the studies used a heterogeneous group for assessing patients with cleft lip and palate (CLP) and, as well as including individuals with unilateral cleft lip and palate (UCLP), also included individuals with unilateral cleft lip (UCL) (Meyer-Marcotty and Stellzig-Eisenhauer, 2009), bilateral cleft lip and palate (BCLP) (Chung et al., 2013; Foo et al., 2013), and cleft palate (CP), cleft lip (CL), and submucous cleft palate (Foo et al., 2013). The remaining seven studies were homogeneous and included only patients with UCLP (Eliason et al., 1991; Al-Omari et al., 2003; Prahl et al., 2006; Papamanou et al., 2012; Gkantidis et al., 2013; Offert et al., 2013; Eichenberger et al., 2014). The number of patients varied from 12 (Papamanou et al., 2012; Gkantidis et al., 2013) to 80 (Foo et al., 2013), and ages ranged from infant to adult. Raters Laypeople The composition of the lay panel was highly variable in both number and demographic profile ranging from four individuals (Foo et al., 2013) to 121 individuals (Chung et al., 2013). Most studies reported using ‘‘laypeople,’’ but some studies also included patients with clefts (Foo et al., 2013; Gkantidis et al., 2013), parents of patients with clefts (Gkantidis et al., 2013), and raters from a medical background (Eliason et al., 1991; Chung et al., 2013) in the laypeople group.
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TABLE 3
Extracted Information From the Included Papers Patients
Author (Year)
Number and Type of Cleft
Comparator Laypeople Demographics and Number
Professional Demographics and Number 42 Professionals (orthodontists, pediatric dentists, general dentists, oral maxillofacial surgeons, craniofacial geneticists, craniofacial plastic surgeons, and otolaryngologists) 24 Professionals (14 orthodontists and 10 maxillofacial surgeons)
Chung et al. (2013)
17 CLP adults corrected by orthognathic surgery; 17 CLP adults corrected by late maxillary protection
121 Laypeople (registered nurses, anesthesia nurses, technicians, front and back office staff, and administrative staff)
Eichenberger et al. (2014)
20 UCLP adults, 10 control adult patients with a Class I occlusion 29 UCLP early-grafted children, 30 UCLP nongrafted children
15 Laypeople
14 Laypeople
5 Professionals (members of the cleft team)
Gkantidis et al. (2013)
12 UCLP young adults
12 Professionals (6 orthodontists, 6 maxillofacial surgeons)
Foo et al. (2013)
80 CLP adults
48 Laypeople (12 adult laypeople matched for patients, 12 adult laypeople matched for parents, 12 patients themselves, 12 patients’ parents) 4 Laypeople (2 laypeople with cleft, 2 laypeople without cleft)
Papamanou et al. (2012)
12 UCLP adults
12 Adult laypeople
Meyer-Marcotty and Stellzig-Eisenhauer (2009)
30 UCL and UCLP adults
15 Laypeople
20 Professionals (10 Orthodontists, 10 oral and maxillofacial surgeons)
Prahl et al. (2006)
41 UCLP infants
24 Laypeople
21 Professionals (members of the cleft team)
Al-Omari et al. (2003)
31 UCLP adolescents and adults
5 Laypeople (4 postgraduate students, 1 schoolteacher)
5 Professionals (1 plastic surgeon, 2 orthodontists, 1 oral maxillofacial surgeon, 1 speech pathologist)
Lo et al. (2002)
64 BCLP children, adolescents, and adults
5 Laypeople
5 Professionals (cleft surgeons)
Eliason et al. (1991)
24 UCLP adolescents and young adults
40 Laypeople (unfamiliar raters included faculty and secretarial staff in other medical department, psychology graduate students, nursing and medical students)
40 Professionals (9 speech pathologists, 6 psychologists, 3 pediatricians, 3 nurses, 3 geneticists, 2 orthodontists, 1 social worker, 13 medical and dental residents)
Offert et al. (2013)
Professionals Studies could be divided into those that grouped raters according to specialty (Lo et al., 2002; MeyerMarcotty and Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Gkantidis et al., 2013; Eichenberger et al., 2014) or those that grouped raters into interdisciplinary care teams (Eliason et al., 1991; Al-Omari et al., 2003; Prahl et al., 2006; Chung et al., 2013; Foo et al., 2013; Offert et al., 2013). Four of the studies based on specialty subdivided the group into orthodontists and surgeons, while one study used a group of professionals comprised of surgeons only (Lo et al., 2002).
5 Professionals: 3 nonsurgical (orthodontist, dentist, psychologist), 2 surgical (plastic surgeons) 24 Professionals (12 orthodontists, 12 maxillofacial surgeons)
However, only three studies went on to compare the rater scores between the two professional groups (Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Eichenberger et al., 2014), while one combined them into a ‘‘single’’ professional group (Gkantidis et al., 2013). The number of professional raters ranged from five (Lo et al., 2002) to 42 (Chung et al., 2013). Type of View In addition to 2D frontal photographs, lateral profile photographs, three-quarter views, worm’s eye, bird’s
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TABLE 3
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Extended Assessment
Type of Image Viewed
Rating Method
Intraoperator
Outcomes Interoperator
Results
Full frontal, full frontal smiling, profile
10-Point scale: 1 - very unattractive, 10 - very attractive
Cronbach a . 0.95
ANOVA mean score
Professionals more critical than laypeople
Frontal, left lateral
100 mm VAS: 0 - very unattractive, 10 - very attractive 5-Point scale (AsherMcDade): 1 - very good appearance, 5 - very poor appearance 100 mm VAS: 0 - not satisfied, 100 - totally satisfied
Linear regression model
Linear regression model; mean VAS score
Laypeople more critical than professionals
Cronbach a . 0.9
Spearman correlation coefficient
Professionals more critical than laypeople
Cronbach a . 0.8
Spearman correlation coefficient
Laypeople more critical than professionals
Frontal left/right profile
100 mm VAS with higher scores reflecting ‘‘very attractive’’
None
Student t test, mean VAS score
Professionals more critical than laypeople
Frontal right/left lateral 3/4 right/lateral 3D
100 mm VAS: 0 - not satisfied, 100 - totally satisfied 9-Point scale: 1 - very asymmetrical or ugly, 9 - very symmetrical or attractive 100 mm VAS, more right position more attractive
Cronbach a . 0.8
Spearman correlation coefficient
Laypeople more critical than professionals
None
Spearman correlation coefficient
Laypeople and professionals rated similarly
Cronbach a
Z-score and ANOVA, mean VAS score
Laypeople and professionals rated similarly
2D, 3D, and clinical assessment, frontal, right and left profile, close-up view of nasolabial and submental area
5-Point scale: 1 - very good appearance, 5 - very poor appearance
Kappa score
Kappa score
Full-face, frontal, lateral, worm’s eye views
3-Point scale: 3 - Good, 2 - Fair, 1 - Poor 6-Point scale: 1 - excellent facial appearance, 6 - much worse than average appearance
Kappa score
Student t test, mean score
Professionals were more critical in rating full face from clinical assessment; laypeople rated full face more critically when using 3D images Laypeople and professionals rated similarly
None
ANOVA and Student t test, mean score
Full face, cropped images of frontal and profile, inferior view of nose Frontal, right and left lateral, 3/4 right lateral
Full-face cropped photos
Frontal and lateral
eye, and cropped images were viewed. Two studies used 3D images (Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009), and one study rated the actual patient (Al-Omari et al., 2003). Rating Method Only one study provided instructions to the panel on how to rate the images (Eichenberger et al., 2014). A second study provided a ‘‘reference’’ image that the raters used as a benchmark for comparison (Prahl et al., 2006). Two studies used a preassessment practice or calibration session prior to the main study to standard-
Professionals more critical than laypeople
ize the rater’s responses (Al-Omari et al., 2003; Offert et al., 2013). Five studies used a VAS (Prahl et al., 2006; Papamanou et al., 2012; Foo et al., 2013; Gkantidis et al., 2013; Eichenberger et al., 2014), and the remaining studies used a categorical scale (Eliason et al., 1991; Lo et al., 2002; Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Offert et al., 2013). All VAS scales ranged from 0 to 100 mm, while categorical scores ranged from five points (AlOmari et al., 2003; Offert et al., 2013), based on the Asher-McDade et al. (1991) scale, to 10 points (Chung et al., 2013).
Moderate High High Moderate Moderate 8 11 11 10 8 1 1 1 1 1 0 1 1 1 0 * Intraoperator assessment refers to the assessment for level of agreement among individual raters within one rater panel. † Interoperator assessment refers to the assessment for level of agreement between rater panels.
0 0 1 0 0 1 2 1 1 1 2 2 2 2 2 1 2 2 2 2 1 1 1 1 1
2 2 2 2 1
Moderate Moderate Moderate High Moderate High 8 10 10 11 9 11 1 1 1 1 1 1 1 0 1 1 0 1 0 0 0 0 0 0 1 2 1 2 2 2 2 2 2 2 2 2 1 2 2 2 2 2 1 2 2 2 1 2
Quality Author (Year)
1 1 1 1 1 1
Total Score Assessors, Professional
Assessment of Study Quality
Interoperator and Intraoperator Assessment
Chung et al. (2013) Eichenberger et al. (2014) Offert et al. (2013) Gkantidis et al. (2013) Foo et al. (2013) Papamanou et al. (2012) Meyer-Marcotty and Stellzig-Eisenhauer (2009) Prahl et al. (2006) Al-Omar et al. (2003) Lo et al. (2002) Eliason et al. (1991)
Full Face Photos 0 No, 1 Yes
Cleft Patients 1 Heterogeneous, 2 Homogeneous
Assessors, Laypeople 1 Medical, 2 Nonmedical
Rating System 1 Categorical Scale, 2 VAS Score, 0–100 mm
Images Viewed Twice 0 No, 1 Yes
Intraoperator Assessment* 0 No, 1 Yes
Interoperator Assessment† 0 No, 1 Yes
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TABLE 4
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In these studies, the intraoperator assessment referred to the level of agreement of individuals in each of the two groups, i.e., professionals and laypeople. Five studies used Cronbach alpha (Prahl et al., 2006; Papamanou et al., 2012; Chung et al., 2013; Gkantidis et al., 2013; Offert et al., 2013), one used a linear regression model (Eichenberger et al., 2014), two used kappa statistics (Lo et al., 2002; Al-Omari et al., 2003), and three did not report any intraoperator assessment (Eliason et al., 1991; Meyer-Marcotty and StellzigEisenhauer, 2009; Foo et al., 2013). Interestingly, one study assessed agreement by rating duplicate images on the same occasion (Eichenberger et al., 2014), and one study repeated the entire rating process (Al-Omari et al., 2003). Interoperator assessment is referred to as the level of agreement between rater panels, i.e., professionals and laypeople. Four studies reported using the Spearman correlation coefficient (Meyer-Marcotty and StellzigEisenhauer, 2009; Papamanou et al., 2012; Gkantidis et al., 2013; Offert et al., 2013), three studies used analysis of variance (ANOVA) (Eliason et al., 1991; Prahl et al., 2006; Chung et al., 2013), two used t tests (Lo et al., 2002; Foo et al., 2013), one used a linear regression model (Eichenberger et al., 2014), and one used kappa statistics (Al-Omari et al., 2003). The interoperator and intraoperator reliability was high for all studies except two in which the intraoperator agreement was fair (Lo et al., 2002) or not stated (Eliason et al., 1991). The individual rater reliability was not assessed in the majority of studies. Only one study reported on the reliability of each rater assessing the facial appearance of patients with cleft lip and palate on two separate occasions (Al-Omari et al., 2003). Three of the 11 publications found that laypeople were more critical than professionals, including two high-level and one moderate-level studies; three studies proposed that there was no significant difference between laypeople and professionals based on one high-level and two moderate-level studies, and five reported that professionals were more critical than laypeople when assessing the facial appearance of patients with cleft lip and palate based on one highlevel and four moderate-level studies. DISCUSSION Numerous studies have reported on the outcome of facial appearance of patients with repaired cleft lip and palate based on the opinion of laypeople and professionals. However, there is no consensus whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial
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FIGURE 1
193
PRISMA flow diagram of study selection process.
images. This may be attributed to the various methodologies and outcome measures used in each of these studies. The present systematic review was performed to determine whether there is a difference between laypeople and professionals in evaluating facial appearance. Viewing Media Four methods have been used to evaluate full facial appearance following cleft lip and palate repair (Sharma et al., 2012): direct clinical assessment (Al-Omari et al., 2003), 2D photography (Eichenberger et al., 2014), 3D imaging (Meyer-Marcotty and Stellzig-Eisenhauer, 2009), and video-graphic assessment (Morrant and Shaw, 1996). Only one study reported on the equivalency of rating facial appearance outcome by direct clinical assessment, 2D photography, and 3D imaging by professionals and laypeople (Al-Omari et al., 2003). The study concluded that there was a statistical difference in the outcome score (modified AsherMcDade) (Asher-McDade et al., 1991) between professionals and laypeople following direct clinical assessment and rating of the full facial 2D images; professionals consistently scored higher indicating a poorer outcome. However, when 3D images were viewed, laypeople gave a poorer outcome score but this was not statistically significant. This single high-quality
study indicates that the viewing media may have an effect on the perceived outcome of the repair. One possible explanation put forward was that laypeople are not trained to critically assess the outcome of cleft lip surgery clinically or in 2D; this together with a degree of sympathy toward the patient may lead to a lower outcome score. Interestingly, in 3D there was no statistically significant difference in outcome score between laypeople and professionals; a possible explanation is that both groups of raters were unfamiliar with viewing 3D images and hence gave similar scores. Cleft Group and Rater Group Selection The inclusion criteria of patients with clefts and laypeople also varied between studies. The homogeneity of cleft patients was not present in some studies (MeyerMarcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Foo et al., 2013). It has been reported that facial growth is different in patients with bilateral cleft lip and palate than in those with unilateral cleft lip and palate (David et al., 2011). This may indicate that different cleft types could affect the result of the final outcome, and heterogeneity in case selection should be avoided (Anderson et al., 2013). The age range of patients with clefts in each study also varied, ranging from infants (Prahl et al., 2006) to adults (Meyer-Marcotty and
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Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Chung et al., 2013; Foo et al., 2013; Eichenberger et al., 2014). The difference between rating infants and adults may result from the various responses to babyishness and cuteness among raters (Hildebrandt and Fitzgerald, 1978). Another potential source of bias is the inclusion of individuals with a medical background in the panel of laypeople (Eliason et al., 1991; Chung et al., 2013). These individuals may have previous knowledge or exposure to cleft patients and may not represent the valid opinion of laypeople (Chung et al., 2013). Thus, for future studies, the homogeneity of both cleft patients and laypeople with a nonmedical background is recommended. The largest discrepancy between studies is the selection of professionals. One opinion is that raters from different professions could differ in the evaluation of facial esthetics in patients with cleft lip and palate (Eichenberger et al., 2014). The professionals should be divided into separate groups according to their disciplines in order to reduce the professional bias. The other opinion is that different specialists in the panel of professionals could represent the professional opinion (Gkantidis et al., 2013). Since the aim of present systematic review is to identify the difference between professionals and laypeople, different specialists were regarded as a whole and included in the professional panel. Besides laypeople and professionals, two studies included patients with cleft lip and palate and their parents in the rater group (Foo et al., 2013; Gkantidis et al., 2013). This is of importance because patientcentered healthcare requires health professionals, patients, their families, and friends to cooperate as a ‘‘treatment team’’ (Lutz and Bowers, 2000). However, patients with cleft lip and palate and their parents may not be regarded as laypeople. It has been reported that professionals, as well as cleft patients and their parents, were less critical than laypeople in assessing facial appearance (Gkantidis et al., 2013). Therefore, in future studies it may be necessary to exclude patients and parents of cleft patients from the laypeople group and view them as a separate assessment panel. Control Group The use of a noncleft control group would determine if professionals and laypeople rated facial appearance similarly and provide a baseline measure. However, only one study used a noncleft control group and found that professionals rated facial appearance higher than laypeople when assessing the full face of individuals (Eichenberger et al., 2014). None of the remaining studies recruited individuals without clefts as a control group. This could be attributed to the aims of the
studies, which were to identify the difference between laypeople and professionals when assessing patients with cleft lip and palate, rather than assessing individuals without clefts. Outcome Measure For outcome measure, the VAS and categorical scales were commonly used by researchers. It was found that the categorical scale seemed to be more discriminative than the VAS (Prahl et al., 2006). However, there was no standard categorical scale of 3, 5, 6, 9, and 10 points applied in the studies (Eliason et al., 1991; Lo et al., 2002; Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Offert et al., 2013). The VAS scores have been shown to be more reliable, valid, and sensitive than the categorical scale, such as the Likert scale, especially for scar rating (Grant et al., 1999; Duncan et al., 2006). Even though VAS scores and Likert scales are presented in metric variables, which allow quantitative evaluations (Gkantidis et al., 2013), VAS is often preferred over categorical scales. Since both VAS and categorical scale are subjective rating systems, reliability of these ratings should be identified. Viewing facial appearance on two separate occasions could determine the reliability of ratings given by each individual rater. Similarly, intraoperator assessment should be performed to assure the agreement within a panel of raters. Interoperator assessment is carried out to investigate the agreement among different groups of raters. Only one study investigated the reliability of viewing the images on two separate occasions (Al-Omari et al., 2003). Following this systematic review, 11 articles were classified according to different levels of evidence quality. Laypeople were found to be more critical than professionals in two high-level and one moderate-level study (Papamanou et al., 2012; Gkantidis et al., 2013). It was inferred that professionals were more familiar with the esthetic outcomes and difficulties of treating patients with cleft lip and palate, and rated less critically than laypeople (Eichenberger et al., 2014). In addition, one high-quality paper and two moderate-quality studies reported no difference between laypeople and professionals (Al-Omari et al., 2003; Prahl et al., 2006). The remaining studies, one high-quality paper and four moderate-quality studies, found that professionals rated more critically than laypeople. Direct comparison of the different studies is difficult due to the difference in viewing media, the composition of the rater group, and the outcome measure. The detailed recommendations for the study design and outcome measure are listed in Table 5. These recommendations will be helpful in investigating rating differences of facial appearance between laypeople and
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TABLE 5
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Recommendations for Study Design and Measurements Recommendations
I. Study design A. Selection of images B. Diagnosis of patients with clefts C. Laypeople D. Professionals E. View the images II. Study measurements A. Subjective rating system B. Intraoperator agreement C. Interoperator agreement
Clinical assessment, 2D photographs, or 3D imaging which include full frontal assessment/views of the face Only one type of patients with clefts could be included to guarantee homogeneity Those from a medical background, previous knowledge of patients with clefts, parents of patients with clefts, and individuals with cleft should not be included Different specialists could be regarded as a whole to represent the professional opinion Raters should view the images on two separate occasions after calibration VAS is preferred, and mean and median of VAS scores of full frontal face should be recorded The level of agreement between rater panels should be performed The level of agreement among individual raters within one rater panel should be performed
professionals in future studies. For example, full facial imaging of a homogeneous group of cleft patients of a similar age should be the target subjects for assessment. One rater panel should be laypeople with no prior knowledge of patients with clefts and no medical background. The other panel should be professionals, even from different disciplines, but who are part of an interdisciplinary care team. Both professionals and laypeople should assess the overall facial appearance of patients with cleft lip and palate using VAS scores on two separate occasions after detailed viewing instructions and calibration. An intraoperator and interoperator reliability should also be performed. The clinical impact of a rating difference between laypeople and professionals could have an influence on the decision for secondary surgical procedures for patients with cleft lip and palate. One possibility is that laypeople and professionals assess the facial appearance similarly, and both agree further surgical intervention would be beneficial. The other possibility is that laypeople and professionals rate facial appearance differently. If laypeople are more critical, then the surgical team may need to manage expectations or discuss the possibility of further surgery. If, however, professionals are more critical, then the facial appearance outcome should be accepted even though the professional may feel the result is suboptimal and can be improved. In summary, this review highlights the need for standardized viewing media that is valid, i.e., records the ‘‘true’’ clinical facial appearance of the individual, composition of a standardized rater panel, and a standardized validated outcome measure. There are apparently many definitions of ‘‘patient-centered care,’’ but the Institute of Medicine definition is thought to be the most acceptable (Mills et al., 2014): ‘‘providing care that is respectful of and responsive to individual patient preferences, needs, and values and ensuring that patient values guide all clinical decisions’’ (Institute of Medicine, 2001). This highlights the importance of including laypeople or the cleft patients peers as judges in the
assessment of soft tissue facial outcome following surgical repair. CONCLUSIONS It still remains unknown whether laypeople are more or less critical than professionals when rating the facial outcome of patients with repaired cleft lip and palate. Professionals are more familiar with the possible esthetic outcomes and difficulties of treating patients with cleft lip and palate. The opposite may be true for laypeople; this disparity between what is achievable by the profession and what is expected by the laypeople may be a source of dissatisfaction in the outcome of facial appearance. Further well-designed studies should be carried out to address this question. REFERENCES Al-Omari I, Millett DT, Ayoub A, Bock M, Ray A, Dunaway D, Crampin L. An appraisal of three methods of rating facial deformity in patients with repaired complete unilateral cleft lip and palate. Cleft Palate Craniofac J. 2003;40:530–537. Anderson NK, Jayaratne YS, Zwahlen RA. Facial aesthetics and perceived need for further treatment among adults with repaired cleft as assessed by cleft team professionals and laypersons. Eur J Orthod. 2013;35:849. Asher-McDade C, Roberts C, Shaw WC, Gallager C. Development of a method for rating nasolabial appearance in patients with clefts of the lip and palate. Cleft Palate Craniofac J. 1991;28:385–390; discussion 390–391. ´ MT, Maia Baratieri C, Alves M Jr, de Souza MM, de Souza Araujo LC. Does rapid maxillary expansion have long-term effects on airway dimensions and breathing? Am J Orthod Dentofacial Orthop. 2011;140:146–156. Bongaarts CA, Prahl-Andersen B, Bronkhorst EM, Spauwen PH, Mulder JW, Vaandrager JM, Kuijpers-Jagtman AM. Effect of infant orthopedics on facial appearance of toddlers with complete unilateral cleft lip and palate (Dutchcleft). Cleft Palate Craniofac J. 2008;45:407–413. Chung EH, Borzabadi-Farahani A, Yen SL. Clinicians and laypeople assessment of facial attractiveness in patients with cleft lip and palate treated with LeFort I surgery or late maxillary protraction. Int J Pediatr Otorhinolaryngol. 2013;77:1446–1450.
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Coetzee V, Greeff JM, Stephen ID, Perrett DI. Cross-cultural agreement in facial attractiveness preferences: the role of ethnicity and gender. PLoS One. 2014;9:e99629. Dai L, Zhu J, Mao M, Li Y, Deng Y, Wang Y, Liang J, Tang L, Wang H, Kilfoy BA, et al. Time trends in oral clefts in Chinese Newborns: data from the Chinese National Birth Defects Monitoring Network. Birth Defects Res A Clin Mol Teratol. 2010;88:41–47. David DJ, Smith I, Nugent M, Richards C, Anderson PJ. From birth to maturity: a group of patients who have completed their protocol management. Part III. Bilateral cleft lip-cleft palate. Plast Reconstr Surg. 2011;128:475–484. Duncan JA, Bond JS, Mason T, Ludlow A, Cridland P, O’Kane S, Ferguson MW. Visual analogue scale scoring and ranking: a suitable and sensitive method for assessing scar quality? Plast Reconstr Surg. 2006;118:909–918. Eichenberger M, Staudt CB, Pandis N, Gnoinski W, Eliades T. Facial attractiveness of patients with unilateral cleft lip and palate and of controls assessed by laypersons and professionals. Eur J Orthod. 2014;36:284–289. Eliason MJ, Hardin MA, Olin WH. Factors that influence ratings of facial appearance for children with cleft lip and palate. Cleft Palate Craniofac J. 1991;28:190–193; discussion 193–194. Foo P, Sampson W, Roberts R, Jamieson L, David D. Facial aesthetics and perceived need for further treatment among adults with repaired cleft as assessed by cleft team professionals and laypersons. Eur J Orthod. 2013;35:341–346. Gkantidis N, Papamanou DA, Christou P, Topouzelis N. Aesthetic outcome of cleft lip and palate treatment. Perceptions of patients, families, and health professionals compared to the general public. J Craniomaxillofac Surg. 2013;41:e105–e110. Grant S, Aitchison T, Henderson E, Christie J, Zare S, McMurray J, Dargie H. A comparison of the reproducibility and the sensitivity to change of visual analogue scales, Borg scales, and Likert scales in normal subjects during submaximal exercise. Chest. 1999;116:1208– 1217. Hildebrandt KA, Fitzgerald HE. Adults’ responses to infants varying in perceived cuteness. Behav Processes. 1978;3:159–172. Institute of Medicine. Crossing the Quality Chasm: A New Health System for the 21st Century / Committee on Quality Health Care in America, Institute of Medicine. Washington, DC: National Academy Press; 2001:40. Kim JB, Strike P, Cadier MC. A simple assessment method for auditing multi-centre unilateral cleft lip repairs. J Plast Reconstr Aesthet Surg. 2011;64:195–200. Kim SC, Nam KC, Rah DK, Cha EJ, Kim DW. Assessment of the cleft nasal deformity using a regression equation. Cleft Palate Craniofac J. 2009;46:197–203. Lagravere MO, Major PW, Flores-Mir C. Long-term dental arch changes after rapid maxillary expansion treatment: a systematic review. Angle Orthod. 2005;75:155–161. Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gøtzsche PC, Ioannidis JP, Clarke M, Devereaux PJ, Kleijnen J, Moher D. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. BMJ. 2009;339:b2700. Little AC, Burriss RP, Jones BC, Roberts SC. Facial appearance affects voting decisions. Evol Hum Behav. 2007;28:18–27. Lo LJ, Wong FH, Mardini S, Chen YR, Noordhoff MS. Assessment of bilateral cleft lip nose deformity- a comparison of results as
judged by cleft surgeons and laypersons. Plast Reconstr Surg. 2002;110:733–738. Lutz BJ, Bowers BJ. Patient-centered care: understanding its interpretation and implementation in health care. Sch Inq Nurs Pract. 2000;14:165–183. Mani MR, Semb G, Andlin-Sobocki A. Nasolabial appearance in adults with repaired unilateral cleft lip and palate: relation between professional and lay rating and patients’ satisfaction. J Plast Surg Hand Surg. 2010;44:191–198. Meyer-Marcotty P, Stellzig-Eisenhauer A. Dentofacial self-perception and social perception of adults with unilateral cleft lip and palate. J Orofac Orthop. 2009;70:224–236. Mills I, Frost J, Cooper C, Moles DR, Kay E. Patient-centered care in general dental practice – a systematic review of the literature. BMC Oral Health. 2014;14:64. Morrant DG, Shaw WC. Use of standardized video recordings to assess cleft surgery outcome. Cleft Palate Craniofac J. 1996;33:134– 142. Moss T. Individual variation in adjusting to visible differences. In: Lansdown R, Rumsey N, Bradbury E, Carr T, Partridge J, eds. Visibly Different. Coping With Disfigurement. Oxford: ButterworthHeinemann; 1997:121–130. Offert B, Janiszewska-Olszowska J, Dudkiewicz Z, Brudnicki A, Katsaros C, Fudalej PS. Facial esthetics in children with unilateral cleft lip and palate 3 years after alveolar bonegrafting combined with rhinoplasty between 2 and 4 years of age. Orthod Craniofac Res. 2013;16:36–43. Papamanou DA, Gkantidis N, Topouzelis N, Christou P. Appreciation of cleft lip and palate treatment outcome by professionals and laypeople. Eur J Orthod. 2012;34:553–560. Pedersen S, Vitaro F, Barker ED, Borge AI. The timing of middlechildhood peer rejection and friendship: linking early behavior to early-adolescent adjustment. Child Dev. 2007;78:1037–1051. Prahl C, Prahl-Andersen B, van’t Hof MA, Kuijpers-Jagtman AM. Infant orthopedics and facial appearance: a randomized clinical trial (Dutchcleft). Cleft Palate Craniofac J. 2006;43:659–664. Rankin M, Borah GL. Perceived functional impact of abnormal facial appearance. Plast Reconstr Surg. 2003;111:2140–2146; discussion 2147–2148. Sarwer DB, Bartlett SP, Whitaker LA, Paige KT, Pertschuk MJ, Wadden TA. Adult psychological functioning of individuals born with craniofacial anomalies. Plast Reconstr Surg. 1999;2:412–418. Sharma VP, Bella H, Cadier MM, Pigott RW, Goodacre TE, Richard BM. Outcomes in facial aesthetics in cleft lip and palate surgery: a systematic review. J Plast Reconstr Aesthet Surg. 2012;65:1233– 1245. Shaw W. Global strategies to reduce the health care burden of craniofacial anomalies: report of WHO meetings on International Collaborative Research on craniofacial anomalies. Cleft Palate Craniofac J. 2004;41:238–243. ´ Stec M, Szczepanska J, Pypec´ J, Hirschfelder U. Periodontal status and oral hygiene in two populations of cleft patients. Cleft Palate Craniofac J. 2007;44:73–78. Tanaka SA, Mahabir RC, Jupiter DC, Menezes JM. Updating the epidemiology of cleft lip with or without cleft palate. Plast Reconstr Surg. 2012;129:511e–518e. Williamson C. The challenge of lay partnership. It provides a different view of the world. BMJ. 1999;319:721–722.
ORIGINAL ARTICLE Evaluation of Facial Appearance in Patients With Cleft Lip and Palate by Laypeople and Professionals: A Systematic Literature Review Shiwen Zhu, B.D.S., M.D.S., Jayakumar Jayaraman, B.D.S., Ph.D., M.Paed. R.C.S. (Edin), Balvinder Khambay, F.D.S. R.C.S. (Eng), M.Orth. R.C.S. (Edin), Ph.D. Objective: To identify whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate (CLP) similarly based on viewing full facial images. Design: Several electronic databases were searched. A hand search was performed among reference lists and relevant journals. Studies that assessed the full facial appearance of patients with CLP based on two-dimensional (2D) photographs, 3D images, or clinical examination by laypeople and professionals using a visual analog scale (VAS) or a categorical rating scale were included. Two authors independently assessed articles using methodologic-quality scoring protocol. Results: Eleven articles were included in qualitative synthesis, including four high-level and seven moderate-level papers. Three studies found that laypeople were more critical than professionals, three found there was no significant difference between laypeople and professionals, and five reported that professionals were more critical than laypeople when assessing facial appearance of patients with CLP. Conclusions: It still remains unknown whether laypeople are more or less critical than professionals when rating facial appearance of patients with repaired CLP. Professionals are more familiar with the esthetic outcomes and difficulties of treating patients. The opposite maybe true for laypeople; this disparity between what is achievable by professionals and what is expected by laypeople may be a source of dissatisfaction in facial appearance outcome. Further well-designed studies should be carried out to address this question and the clinical significance of the difference in rating scores for patients with CLP. KEY WORDS:
cleft lip and palate, facial appearance, laypeople, professionals, systematic review
The first of these is a primary lip repair, which is carried out to improve function, speech, and esthetics. Inadequate correction of facial esthetics, in particular nasolabial symmetry is thought to be a source of emotional distress and dissatisfaction among this group of patients (Gkantidis et al., 2013). This may be the reason for further surgical revision procedures (Foo et al., 2013). In the present era of patient-centered interdisciplinary management, in addition to the patient, professionals and laypeople may also indirectly affect the decision for further surgery. Even though the patient’s opinion is paramount in requesting further surgery to improve his or her perceived quality of life (Chung et al., 2013), the opinion and support of laypeople such as friends and peers have been shown to have a positive effect on the psychological well-being of the individual (Moss, 1997; Pedersen et al., 2007). Facial attractiveness is of critical importance in various social interactions (Little et al., 2007; Coetzee et al., 2014). Social interactions of patients primarily depend on perceptions of the wider community, which mainly consists of laypeople or peers (Williamson, 1999; Rankin and Borah, 2003). It has been shown that patients with cleft lip and palate often experience negative social interactions as a result of an unattractive facial appearance (Chung et al.,
Cleft lip and palate is the most common congenital dysmorphology affecting the oral and maxillofacial region (Stec et al., 2007). The prevalence of cleft lip and palate varies between countries, socioeconomic status, and ethnicities (Shaw, 2004). The average worldwide prevalence of patients with cleft lip with or without cleft palate has been reported as 7.94 per 10,000 births (Tanaka et al., 2012). Among the ethnicities, the Chinese have the highest prevalence of cleft lip and palate, affecting 16.63 per 10,000 births during 1996 to 2005, with an upward trend over time (Dai et al., 2010). In order to address the facial dysmorphology of patients with cleft lip and palate, a series of surgical interventions are undertaken at specific times during the child’s development.
S. Zhu and Dr. Jayaraman are research postgraduate students and Dr. Khambay is Clinical Associate Professor, Discipline of Orthodontics and Paediatric Dentistry, Faculty of Dentistry, The University of Hong Kong, Hong Kong, Hong Kong SAR. Submitted June 2014; Revised September 2014, November 2014; Accepted December 2014. Address correspondence to: Dr. Balvinder Khambay, 2A, Prince Philip Dental Hospital, 34 Hospital Road, Hong Kong. E-mail [email protected]. DOI: 10.1597/14-177 187
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2013). Individuals with congenital craniofacial conditions often experience discrimination in an employment and social setting related to lower satisfaction with facial appearance, quality of life, and self-esteem (Sarwer et al., 1999). This highlights the need to involve laypeople as potential peers in assessing facial appearance outcome. Numerous studies have investigated the outcome of postsurgical facial appearance in patients with cleft lip and palate based on the views of laypeople and professionals. Some studies have focused on cropped photographs of the isolated nasolabial region (Bongaarts et al., 2008; Kim et al., 2009, 2011; Mani et al., 2010), while others evaluated the full frontal facial image of patients with cleft lip and palate (Lo et al., 2002; Papamanou et al., 2012; Chung et al., 2013; Gkantidis et al., 2013; Offert et al., 2013). The use of cropped photographs may not be appropriate as they do not indicate total facial harmony and may therefore be misleading. The esthetic outcome of the repaired cleft lip should not be viewed in isolation but should be based on overall facial appearance as it is in other facial dysmorphologies (i.e., orthognathic patients). There is agreement that laypeople and professionals rate the facial appearance of patients with cleft lip and palate consistently lower than noncleft individuals (Eichenberger et al., 2014). However, when assessing the facial appearance of individuals with clefts there are conflicting opinions between laypeople and professionals. Some studies have reported professionals are more critical (Eliason et al., 1991; Chung et al., 2013; Foo et al., 2013; Offert et al., 2013), while others reported that laypeople were more critical (Papamanou et al., 2012; Gkantidis et al., 2013; Eichenberger et al., 2014). The aim of this systematic review was, therefore, to determine whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial images. METHODS Eligibility Criteria The selected studies compared full facial appearance of cleft repair as judged by laypeople and professionals. Studies that met the following criteria were included: (1) patients with cleft lip with or without cleft palate; (2) outcome of facial appearance assessed by laypeople and professionals; (3) outcome measure based on a visual analog scale (VAS) or a categorical rating scale; (4) use of two-dimensional (2D) photographs, 3D images, or real patients; and (5) assessment of full facial images. Exclusion criteria were as follows: (1) patients without cleft lip; (2) only one group of assessors; (3) use of video images; (4) reviews, letter to editors, expert opinions, and case reports; and (5) partial facial images, i.e., cropped or isolated surgical cleft region.
Information Resources PubMed (all available articles until week 2 of June 2014), The Cochrane Library (1999 to week 2 of June 2014), Web of Science (1956 to week 2 of June 2014), Scopus (1999 to week 2 of June 2014), and EMBASE (1980 to week 2 of June 2014) were searched to identify relevant articles. The reference lists in these relevant articles were retrieved manually. The articles in Cleft Palate-Craniofacial Journal (from January 2004 to May 2014) and American Journal of Orthodontics and Dentofacial Orthopedics (from January 2004 to June 2014) were also manually searched based on their relevance to facial appearance and the patients with cleft lip and palate. No language limit was set. Review Question The patients, intervention, comparator, and outcome (PICO) structure was applied to form the research question. In this systematic review, patients are those with cleft lip and palate. The comparison will be carried out between the professionals and laypeople. The outcome will be the difference in evaluation of facial appearance by professionals and laypeople. Therefore, the review question is whether professionals and laypeople rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial images. Search Strategy Search terms were identified to fit the research question, including cleft, laypeople, professionals, assess, face, and appearance. Based on these search terms, the search strategy was as shown in Table 1. PRISMA guidelines were used to conduct the present systematic review (Liberati et al., 2009). Initially, two authors (S.Z. and B.K.) independently searched the databases on the basis of title and abstract. Papers that met the eligibility criteria were included. If the eligibility of papers could not be identified by title and abstract, the full-text articles were read by the reviewers. An additional search was performed manually utilizing the reference lists of included papers and relevant journals for identifying eligible articles. Quality Assessment To evaluate the methodologic quality of each article, an eight criteria-based scoring protocol was used, modified from Lagravere et al. (2005), as shown in Table 2. Two authors independently completed the information extraction sheet for the included papers, which was designed on the basis of the review question and PICO structure, as shown in Table 3. Disagreement between
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TABLE 1
Search Strategy for Electronic Databases
PubMed ((((((cleft) OR harelip)AND (((((laypeople) OR layperson*) OR layrater*) OR non-profession*) OR general public) OR lay public) AND (((((profession*) OR clinician*) OR orthodonti*) OR surgeon*) OR maxillofacial surgeon*) OR oral surgeon* ) AND ((((((assess*) OR evaluat*) OR prefer*) OR perceive) OR perception) OR view) OR psycholog* ) AND (((((face) OR facial) OR dentofacial) OR profile) OR lateral) OR cephalometric*) AND ((((attract*) OR aesthetic*) OR esthetic*) OR appearance) OR beauty Cochrane Library (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) Web of Science (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) Scopus (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty) EMBASE (cleft OR harelip) AND (laypeople OR layperson* OR layrater* or non-profession* OR general public OR lay public) AND (profession* or clinician* or orthodonti* or surgeon* or maxillofacial surgeon* or oral surgeon*) AND (assess* OR evaluat* OR prefer* OR perceive OR perception OR view OR psycholog*) AND (face OR facial OR dentofacial OR profile OR lateral OR cephalometric*) AND ( attract* OR aesthetic* OR esthetic* OR appearance OR beauty)
two authors was resolved by discussion and consensus. When two authors could not reach an agreement, a third reviewer (J.J.) was asked and consensus reached. According to the total score, studies were classified into low (score ,6), moderate (score .6 and ,10), and high (score .10) level of quality, as shown in Table 4 (Lagravere et al., 2005; Baratieri et al., 2011). RESULTS In total, 112 studies were identified following the database searches. Eleven articles were finally included in qualitative synthesis. The retrieval of PubMed, The Cochrane Library, Web of Science, Scopus, and EMBASE provided 19, 5, 18, 58, and 12 records, respectively. After removing duplicates and ineligible articles, 11 publications
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TABLE 2 Methodologic-Quality Scoring Protocol (Maximum Score, 12 Points) Based on Lagravere et al. (2005) I. Study design (8) A. Full face photograph (2D or 3D), including frontal side (1) B. Cleft patients: homogeneous (2) or heterogeneous (1) C. Viewed by professionals (2) D. Viewed by laypeople: nonmedical background (2) or medical background (1) E. Viewed on two separate occasions (1) II. Study measurements (4) A. Rating system: VAS scale 0 to 100 mm (2) or categorical scale (3-, 4-, 5-, 6-, 9-, and 10-point scale) (1) B. Intrapanel agreement (1) C. Interpanel agreement (1)
remained. Seven additional records were identified through reference linkage by hand searching. After reading the fulltext articles, 11 of them were finally included in qualitative synthesis. The PRISMA flow diagram is shown in Figure 1. Number and Type of Patients With Clefts The majority of studies evaluated the facial appearance of unilateral cleft lip and palate; only one study evaluated patients with bilateral cleft lip and palate (Lo et al., 2002). However, three of the studies used a heterogeneous group for assessing patients with cleft lip and palate (CLP) and, as well as including individuals with unilateral cleft lip and palate (UCLP), also included individuals with unilateral cleft lip (UCL) (Meyer-Marcotty and Stellzig-Eisenhauer, 2009), bilateral cleft lip and palate (BCLP) (Chung et al., 2013; Foo et al., 2013), and cleft palate (CP), cleft lip (CL), and submucous cleft palate (Foo et al., 2013). The remaining seven studies were homogeneous and included only patients with UCLP (Eliason et al., 1991; Al-Omari et al., 2003; Prahl et al., 2006; Papamanou et al., 2012; Gkantidis et al., 2013; Offert et al., 2013; Eichenberger et al., 2014). The number of patients varied from 12 (Papamanou et al., 2012; Gkantidis et al., 2013) to 80 (Foo et al., 2013), and ages ranged from infant to adult. Raters Laypeople The composition of the lay panel was highly variable in both number and demographic profile ranging from four individuals (Foo et al., 2013) to 121 individuals (Chung et al., 2013). Most studies reported using ‘‘laypeople,’’ but some studies also included patients with clefts (Foo et al., 2013; Gkantidis et al., 2013), parents of patients with clefts (Gkantidis et al., 2013), and raters from a medical background (Eliason et al., 1991; Chung et al., 2013) in the laypeople group.
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TABLE 3
Extracted Information From the Included Papers Patients
Author (Year)
Number and Type of Cleft
Comparator Laypeople Demographics and Number
Professional Demographics and Number 42 Professionals (orthodontists, pediatric dentists, general dentists, oral maxillofacial surgeons, craniofacial geneticists, craniofacial plastic surgeons, and otolaryngologists) 24 Professionals (14 orthodontists and 10 maxillofacial surgeons)
Chung et al. (2013)
17 CLP adults corrected by orthognathic surgery; 17 CLP adults corrected by late maxillary protection
121 Laypeople (registered nurses, anesthesia nurses, technicians, front and back office staff, and administrative staff)
Eichenberger et al. (2014)
20 UCLP adults, 10 control adult patients with a Class I occlusion 29 UCLP early-grafted children, 30 UCLP nongrafted children
15 Laypeople
14 Laypeople
5 Professionals (members of the cleft team)
Gkantidis et al. (2013)
12 UCLP young adults
12 Professionals (6 orthodontists, 6 maxillofacial surgeons)
Foo et al. (2013)
80 CLP adults
48 Laypeople (12 adult laypeople matched for patients, 12 adult laypeople matched for parents, 12 patients themselves, 12 patients’ parents) 4 Laypeople (2 laypeople with cleft, 2 laypeople without cleft)
Papamanou et al. (2012)
12 UCLP adults
12 Adult laypeople
Meyer-Marcotty and Stellzig-Eisenhauer (2009)
30 UCL and UCLP adults
15 Laypeople
20 Professionals (10 Orthodontists, 10 oral and maxillofacial surgeons)
Prahl et al. (2006)
41 UCLP infants
24 Laypeople
21 Professionals (members of the cleft team)
Al-Omari et al. (2003)
31 UCLP adolescents and adults
5 Laypeople (4 postgraduate students, 1 schoolteacher)
5 Professionals (1 plastic surgeon, 2 orthodontists, 1 oral maxillofacial surgeon, 1 speech pathologist)
Lo et al. (2002)
64 BCLP children, adolescents, and adults
5 Laypeople
5 Professionals (cleft surgeons)
Eliason et al. (1991)
24 UCLP adolescents and young adults
40 Laypeople (unfamiliar raters included faculty and secretarial staff in other medical department, psychology graduate students, nursing and medical students)
40 Professionals (9 speech pathologists, 6 psychologists, 3 pediatricians, 3 nurses, 3 geneticists, 2 orthodontists, 1 social worker, 13 medical and dental residents)
Offert et al. (2013)
Professionals Studies could be divided into those that grouped raters according to specialty (Lo et al., 2002; MeyerMarcotty and Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Gkantidis et al., 2013; Eichenberger et al., 2014) or those that grouped raters into interdisciplinary care teams (Eliason et al., 1991; Al-Omari et al., 2003; Prahl et al., 2006; Chung et al., 2013; Foo et al., 2013; Offert et al., 2013). Four of the studies based on specialty subdivided the group into orthodontists and surgeons, while one study used a group of professionals comprised of surgeons only (Lo et al., 2002).
5 Professionals: 3 nonsurgical (orthodontist, dentist, psychologist), 2 surgical (plastic surgeons) 24 Professionals (12 orthodontists, 12 maxillofacial surgeons)
However, only three studies went on to compare the rater scores between the two professional groups (Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Eichenberger et al., 2014), while one combined them into a ‘‘single’’ professional group (Gkantidis et al., 2013). The number of professional raters ranged from five (Lo et al., 2002) to 42 (Chung et al., 2013). Type of View In addition to 2D frontal photographs, lateral profile photographs, three-quarter views, worm’s eye, bird’s
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Extended Assessment
Type of Image Viewed
Rating Method
Intraoperator
Outcomes Interoperator
Results
Full frontal, full frontal smiling, profile
10-Point scale: 1 - very unattractive, 10 - very attractive
Cronbach a . 0.95
ANOVA mean score
Professionals more critical than laypeople
Frontal, left lateral
100 mm VAS: 0 - very unattractive, 10 - very attractive 5-Point scale (AsherMcDade): 1 - very good appearance, 5 - very poor appearance 100 mm VAS: 0 - not satisfied, 100 - totally satisfied
Linear regression model
Linear regression model; mean VAS score
Laypeople more critical than professionals
Cronbach a . 0.9
Spearman correlation coefficient
Professionals more critical than laypeople
Cronbach a . 0.8
Spearman correlation coefficient
Laypeople more critical than professionals
Frontal left/right profile
100 mm VAS with higher scores reflecting ‘‘very attractive’’
None
Student t test, mean VAS score
Professionals more critical than laypeople
Frontal right/left lateral 3/4 right/lateral 3D
100 mm VAS: 0 - not satisfied, 100 - totally satisfied 9-Point scale: 1 - very asymmetrical or ugly, 9 - very symmetrical or attractive 100 mm VAS, more right position more attractive
Cronbach a . 0.8
Spearman correlation coefficient
Laypeople more critical than professionals
None
Spearman correlation coefficient
Laypeople and professionals rated similarly
Cronbach a
Z-score and ANOVA, mean VAS score
Laypeople and professionals rated similarly
2D, 3D, and clinical assessment, frontal, right and left profile, close-up view of nasolabial and submental area
5-Point scale: 1 - very good appearance, 5 - very poor appearance
Kappa score
Kappa score
Full-face, frontal, lateral, worm’s eye views
3-Point scale: 3 - Good, 2 - Fair, 1 - Poor 6-Point scale: 1 - excellent facial appearance, 6 - much worse than average appearance
Kappa score
Student t test, mean score
Professionals were more critical in rating full face from clinical assessment; laypeople rated full face more critically when using 3D images Laypeople and professionals rated similarly
None
ANOVA and Student t test, mean score
Full face, cropped images of frontal and profile, inferior view of nose Frontal, right and left lateral, 3/4 right lateral
Full-face cropped photos
Frontal and lateral
eye, and cropped images were viewed. Two studies used 3D images (Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009), and one study rated the actual patient (Al-Omari et al., 2003). Rating Method Only one study provided instructions to the panel on how to rate the images (Eichenberger et al., 2014). A second study provided a ‘‘reference’’ image that the raters used as a benchmark for comparison (Prahl et al., 2006). Two studies used a preassessment practice or calibration session prior to the main study to standard-
Professionals more critical than laypeople
ize the rater’s responses (Al-Omari et al., 2003; Offert et al., 2013). Five studies used a VAS (Prahl et al., 2006; Papamanou et al., 2012; Foo et al., 2013; Gkantidis et al., 2013; Eichenberger et al., 2014), and the remaining studies used a categorical scale (Eliason et al., 1991; Lo et al., 2002; Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Offert et al., 2013). All VAS scales ranged from 0 to 100 mm, while categorical scores ranged from five points (AlOmari et al., 2003; Offert et al., 2013), based on the Asher-McDade et al. (1991) scale, to 10 points (Chung et al., 2013).
Moderate High High Moderate Moderate 8 11 11 10 8 1 1 1 1 1 0 1 1 1 0 * Intraoperator assessment refers to the assessment for level of agreement among individual raters within one rater panel. † Interoperator assessment refers to the assessment for level of agreement between rater panels.
0 0 1 0 0 1 2 1 1 1 2 2 2 2 2 1 2 2 2 2 1 1 1 1 1
2 2 2 2 1
Moderate Moderate Moderate High Moderate High 8 10 10 11 9 11 1 1 1 1 1 1 1 0 1 1 0 1 0 0 0 0 0 0 1 2 1 2 2 2 2 2 2 2 2 2 1 2 2 2 2 2 1 2 2 2 1 2
Quality Author (Year)
1 1 1 1 1 1
Total Score Assessors, Professional
Assessment of Study Quality
Interoperator and Intraoperator Assessment
Chung et al. (2013) Eichenberger et al. (2014) Offert et al. (2013) Gkantidis et al. (2013) Foo et al. (2013) Papamanou et al. (2012) Meyer-Marcotty and Stellzig-Eisenhauer (2009) Prahl et al. (2006) Al-Omar et al. (2003) Lo et al. (2002) Eliason et al. (1991)
Full Face Photos 0 No, 1 Yes
Cleft Patients 1 Heterogeneous, 2 Homogeneous
Assessors, Laypeople 1 Medical, 2 Nonmedical
Rating System 1 Categorical Scale, 2 VAS Score, 0–100 mm
Images Viewed Twice 0 No, 1 Yes
Intraoperator Assessment* 0 No, 1 Yes
Interoperator Assessment† 0 No, 1 Yes
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TABLE 4
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In these studies, the intraoperator assessment referred to the level of agreement of individuals in each of the two groups, i.e., professionals and laypeople. Five studies used Cronbach alpha (Prahl et al., 2006; Papamanou et al., 2012; Chung et al., 2013; Gkantidis et al., 2013; Offert et al., 2013), one used a linear regression model (Eichenberger et al., 2014), two used kappa statistics (Lo et al., 2002; Al-Omari et al., 2003), and three did not report any intraoperator assessment (Eliason et al., 1991; Meyer-Marcotty and StellzigEisenhauer, 2009; Foo et al., 2013). Interestingly, one study assessed agreement by rating duplicate images on the same occasion (Eichenberger et al., 2014), and one study repeated the entire rating process (Al-Omari et al., 2003). Interoperator assessment is referred to as the level of agreement between rater panels, i.e., professionals and laypeople. Four studies reported using the Spearman correlation coefficient (Meyer-Marcotty and StellzigEisenhauer, 2009; Papamanou et al., 2012; Gkantidis et al., 2013; Offert et al., 2013), three studies used analysis of variance (ANOVA) (Eliason et al., 1991; Prahl et al., 2006; Chung et al., 2013), two used t tests (Lo et al., 2002; Foo et al., 2013), one used a linear regression model (Eichenberger et al., 2014), and one used kappa statistics (Al-Omari et al., 2003). The interoperator and intraoperator reliability was high for all studies except two in which the intraoperator agreement was fair (Lo et al., 2002) or not stated (Eliason et al., 1991). The individual rater reliability was not assessed in the majority of studies. Only one study reported on the reliability of each rater assessing the facial appearance of patients with cleft lip and palate on two separate occasions (Al-Omari et al., 2003). Three of the 11 publications found that laypeople were more critical than professionals, including two high-level and one moderate-level studies; three studies proposed that there was no significant difference between laypeople and professionals based on one high-level and two moderate-level studies, and five reported that professionals were more critical than laypeople when assessing the facial appearance of patients with cleft lip and palate based on one highlevel and four moderate-level studies. DISCUSSION Numerous studies have reported on the outcome of facial appearance of patients with repaired cleft lip and palate based on the opinion of laypeople and professionals. However, there is no consensus whether laypeople and professionals rate the facial appearance of patients with cleft lip and palate similarly based on viewing full facial
Zhu et al., EVALUATION OF FACIAL APPEARANCE BY LAYPEOPLE AND PROFESSIONALS
FIGURE 1
193
PRISMA flow diagram of study selection process.
images. This may be attributed to the various methodologies and outcome measures used in each of these studies. The present systematic review was performed to determine whether there is a difference between laypeople and professionals in evaluating facial appearance. Viewing Media Four methods have been used to evaluate full facial appearance following cleft lip and palate repair (Sharma et al., 2012): direct clinical assessment (Al-Omari et al., 2003), 2D photography (Eichenberger et al., 2014), 3D imaging (Meyer-Marcotty and Stellzig-Eisenhauer, 2009), and video-graphic assessment (Morrant and Shaw, 1996). Only one study reported on the equivalency of rating facial appearance outcome by direct clinical assessment, 2D photography, and 3D imaging by professionals and laypeople (Al-Omari et al., 2003). The study concluded that there was a statistical difference in the outcome score (modified AsherMcDade) (Asher-McDade et al., 1991) between professionals and laypeople following direct clinical assessment and rating of the full facial 2D images; professionals consistently scored higher indicating a poorer outcome. However, when 3D images were viewed, laypeople gave a poorer outcome score but this was not statistically significant. This single high-quality
study indicates that the viewing media may have an effect on the perceived outcome of the repair. One possible explanation put forward was that laypeople are not trained to critically assess the outcome of cleft lip surgery clinically or in 2D; this together with a degree of sympathy toward the patient may lead to a lower outcome score. Interestingly, in 3D there was no statistically significant difference in outcome score between laypeople and professionals; a possible explanation is that both groups of raters were unfamiliar with viewing 3D images and hence gave similar scores. Cleft Group and Rater Group Selection The inclusion criteria of patients with clefts and laypeople also varied between studies. The homogeneity of cleft patients was not present in some studies (MeyerMarcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Foo et al., 2013). It has been reported that facial growth is different in patients with bilateral cleft lip and palate than in those with unilateral cleft lip and palate (David et al., 2011). This may indicate that different cleft types could affect the result of the final outcome, and heterogeneity in case selection should be avoided (Anderson et al., 2013). The age range of patients with clefts in each study also varied, ranging from infants (Prahl et al., 2006) to adults (Meyer-Marcotty and
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Stellzig-Eisenhauer, 2009; Papamanou et al., 2012; Chung et al., 2013; Foo et al., 2013; Eichenberger et al., 2014). The difference between rating infants and adults may result from the various responses to babyishness and cuteness among raters (Hildebrandt and Fitzgerald, 1978). Another potential source of bias is the inclusion of individuals with a medical background in the panel of laypeople (Eliason et al., 1991; Chung et al., 2013). These individuals may have previous knowledge or exposure to cleft patients and may not represent the valid opinion of laypeople (Chung et al., 2013). Thus, for future studies, the homogeneity of both cleft patients and laypeople with a nonmedical background is recommended. The largest discrepancy between studies is the selection of professionals. One opinion is that raters from different professions could differ in the evaluation of facial esthetics in patients with cleft lip and palate (Eichenberger et al., 2014). The professionals should be divided into separate groups according to their disciplines in order to reduce the professional bias. The other opinion is that different specialists in the panel of professionals could represent the professional opinion (Gkantidis et al., 2013). Since the aim of present systematic review is to identify the difference between professionals and laypeople, different specialists were regarded as a whole and included in the professional panel. Besides laypeople and professionals, two studies included patients with cleft lip and palate and their parents in the rater group (Foo et al., 2013; Gkantidis et al., 2013). This is of importance because patientcentered healthcare requires health professionals, patients, their families, and friends to cooperate as a ‘‘treatment team’’ (Lutz and Bowers, 2000). However, patients with cleft lip and palate and their parents may not be regarded as laypeople. It has been reported that professionals, as well as cleft patients and their parents, were less critical than laypeople in assessing facial appearance (Gkantidis et al., 2013). Therefore, in future studies it may be necessary to exclude patients and parents of cleft patients from the laypeople group and view them as a separate assessment panel. Control Group The use of a noncleft control group would determine if professionals and laypeople rated facial appearance similarly and provide a baseline measure. However, only one study used a noncleft control group and found that professionals rated facial appearance higher than laypeople when assessing the full face of individuals (Eichenberger et al., 2014). None of the remaining studies recruited individuals without clefts as a control group. This could be attributed to the aims of the
studies, which were to identify the difference between laypeople and professionals when assessing patients with cleft lip and palate, rather than assessing individuals without clefts. Outcome Measure For outcome measure, the VAS and categorical scales were commonly used by researchers. It was found that the categorical scale seemed to be more discriminative than the VAS (Prahl et al., 2006). However, there was no standard categorical scale of 3, 5, 6, 9, and 10 points applied in the studies (Eliason et al., 1991; Lo et al., 2002; Al-Omari et al., 2003; Meyer-Marcotty and Stellzig-Eisenhauer, 2009; Chung et al., 2013; Offert et al., 2013). The VAS scores have been shown to be more reliable, valid, and sensitive than the categorical scale, such as the Likert scale, especially for scar rating (Grant et al., 1999; Duncan et al., 2006). Even though VAS scores and Likert scales are presented in metric variables, which allow quantitative evaluations (Gkantidis et al., 2013), VAS is often preferred over categorical scales. Since both VAS and categorical scale are subjective rating systems, reliability of these ratings should be identified. Viewing facial appearance on two separate occasions could determine the reliability of ratings given by each individual rater. Similarly, intraoperator assessment should be performed to assure the agreement within a panel of raters. Interoperator assessment is carried out to investigate the agreement among different groups of raters. Only one study investigated the reliability of viewing the images on two separate occasions (Al-Omari et al., 2003). Following this systematic review, 11 articles were classified according to different levels of evidence quality. Laypeople were found to be more critical than professionals in two high-level and one moderate-level study (Papamanou et al., 2012; Gkantidis et al., 2013). It was inferred that professionals were more familiar with the esthetic outcomes and difficulties of treating patients with cleft lip and palate, and rated less critically than laypeople (Eichenberger et al., 2014). In addition, one high-quality paper and two moderate-quality studies reported no difference between laypeople and professionals (Al-Omari et al., 2003; Prahl et al., 2006). The remaining studies, one high-quality paper and four moderate-quality studies, found that professionals rated more critically than laypeople. Direct comparison of the different studies is difficult due to the difference in viewing media, the composition of the rater group, and the outcome measure. The detailed recommendations for the study design and outcome measure are listed in Table 5. These recommendations will be helpful in investigating rating differences of facial appearance between laypeople and
Zhu et al., EVALUATION OF FACIAL APPEARANCE BY LAYPEOPLE AND PROFESSIONALS
TABLE 5
195
Recommendations for Study Design and Measurements Recommendations
I. Study design A. Selection of images B. Diagnosis of patients with clefts C. Laypeople D. Professionals E. View the images II. Study measurements A. Subjective rating system B. Intraoperator agreement C. Interoperator agreement
Clinical assessment, 2D photographs, or 3D imaging which include full frontal assessment/views of the face Only one type of patients with clefts could be included to guarantee homogeneity Those from a medical background, previous knowledge of patients with clefts, parents of patients with clefts, and individuals with cleft should not be included Different specialists could be regarded as a whole to represent the professional opinion Raters should view the images on two separate occasions after calibration VAS is preferred, and mean and median of VAS scores of full frontal face should be recorded The level of agreement between rater panels should be performed The level of agreement among individual raters within one rater panel should be performed
professionals in future studies. For example, full facial imaging of a homogeneous group of cleft patients of a similar age should be the target subjects for assessment. One rater panel should be laypeople with no prior knowledge of patients with clefts and no medical background. The other panel should be professionals, even from different disciplines, but who are part of an interdisciplinary care team. Both professionals and laypeople should assess the overall facial appearance of patients with cleft lip and palate using VAS scores on two separate occasions after detailed viewing instructions and calibration. An intraoperator and interoperator reliability should also be performed. The clinical impact of a rating difference between laypeople and professionals could have an influence on the decision for secondary surgical procedures for patients with cleft lip and palate. One possibility is that laypeople and professionals assess the facial appearance similarly, and both agree further surgical intervention would be beneficial. The other possibility is that laypeople and professionals rate facial appearance differently. If laypeople are more critical, then the surgical team may need to manage expectations or discuss the possibility of further surgery. If, however, professionals are more critical, then the facial appearance outcome should be accepted even though the professional may feel the result is suboptimal and can be improved. In summary, this review highlights the need for standardized viewing media that is valid, i.e., records the ‘‘true’’ clinical facial appearance of the individual, composition of a standardized rater panel, and a standardized validated outcome measure. There are apparently many definitions of ‘‘patient-centered care,’’ but the Institute of Medicine definition is thought to be the most acceptable (Mills et al., 2014): ‘‘providing care that is respectful of and responsive to individual patient preferences, needs, and values and ensuring that patient values guide all clinical decisions’’ (Institute of Medicine, 2001). This highlights the importance of including laypeople or the cleft patients peers as judges in the
assessment of soft tissue facial outcome following surgical repair. CONCLUSIONS It still remains unknown whether laypeople are more or less critical than professionals when rating the facial outcome of patients with repaired cleft lip and palate. Professionals are more familiar with the possible esthetic outcomes and difficulties of treating patients with cleft lip and palate. The opposite may be true for laypeople; this disparity between what is achievable by the profession and what is expected by the laypeople may be a source of dissatisfaction in the outcome of facial appearance. Further well-designed studies should be carried out to address this question. REFERENCES Al-Omari I, Millett DT, Ayoub A, Bock M, Ray A, Dunaway D, Crampin L. An appraisal of three methods of rating facial deformity in patients with repaired complete unilateral cleft lip and palate. Cleft Palate Craniofac J. 2003;40:530–537. Anderson NK, Jayaratne YS, Zwahlen RA. Facial aesthetics and perceived need for further treatment among adults with repaired cleft as assessed by cleft team professionals and laypersons. Eur J Orthod. 2013;35:849. Asher-McDade C, Roberts C, Shaw WC, Gallager C. Development of a method for rating nasolabial appearance in patients with clefts of the lip and palate. Cleft Palate Craniofac J. 1991;28:385–390; discussion 390–391. ´ MT, Maia Baratieri C, Alves M Jr, de Souza MM, de Souza Araujo LC. Does rapid maxillary expansion have long-term effects on airway dimensions and breathing? Am J Orthod Dentofacial Orthop. 2011;140:146–156. Bongaarts CA, Prahl-Andersen B, Bronkhorst EM, Spauwen PH, Mulder JW, Vaandrager JM, Kuijpers-Jagtman AM. Effect of infant orthopedics on facial appearance of toddlers with complete unilateral cleft lip and palate (Dutchcleft). Cleft Palate Craniofac J. 2008;45:407–413. Chung EH, Borzabadi-Farahani A, Yen SL. Clinicians and laypeople assessment of facial attractiveness in patients with cleft lip and palate treated with LeFort I surgery or late maxillary protraction. Int J Pediatr Otorhinolaryngol. 2013;77:1446–1450.
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