Original Article

The Cleft Palate-Craniofacial Journal 1-7 ª 2020, American Cleft Palate- Comparing Quality of Life and Perceptual Craniofacial Association Article reuse guidelines: Speech Ratings in Children With Cleft sagepub.com/journals-permissions DOI: 10.1177/1055665620949435 Palate journals.sagepub.com/home/cpc

Hailey M. Pedersen, MS, CCC-SLP1, Paige A. Goodie, MS, CCC-SLP2, Maia N. Braden, MS, CCC-SLP1, and Susan L. Thibeault, MS, PhD, CCC-SLP1

Abstract Objective: To delineate the relationship between patient and parent-reported quality of life (QOL) ratings and perceptual characteristics of speech assigned by a speech-language pathologist (SLP) in children with repaired cleft palate. Design: Prospective. Setting: Academic Children’s Hospital. Participants: This population-based sample included children, aged 3 to 18 with a history of repaired cleft palate, and their parents. Intervention: Participants completed the Velopharyngeal Insufficiency Effects on Life Outcomes Questionnaire (VELO). Children’s speech was judged perceptually by an expert SLP using the Pittsburgh Weighted Speech Scale (PWSS). Main Outcome Measure(s): Velopharyngeal Insufficiency Effects on Life Outcomes questionnaire assessed participant and parent perceptions of impact of velopharyngeal function on QOL. Pittsburgh Weighted Speech Scale assessed nasal emissions, facial grimacing, nasality, quality of phonation, and articulation. Results: Enrollment included 48 participant parent dyads. Overall, participants reported high QOL scores within the 95% CI with children reporting slightly better yet not significantly different QOL (86.27 + 8.96) compared to their parents (81.81 + 15.2). Children received an average score of 1.38 + 1.96 on the PWSS corresponding to borderline velopharyngeal com- petence. A significant moderate negative correlation was found between PWSS total score and parent VELO total score (r ¼0.51103, P ¼ .0002). Mild–moderate significant negative correlations were measured between PWSS total and the 5 subscales of the VELO. No significant correlations were measured between PWSS and child VELO total responses or between total scores and subscales. Conclusions: Results suggest that as perceptual analysis of speech improves, overall QOL improves moderately.

Keywords cleft palate, quality of life, speech

Introduction congenital anomalies of the face (Kummer et al., 2012). Treat- ment for cleft lip/palate is surgical with the primary goal for Children with cleft lip/palate can have a range of communica- tion disorders, which have distinct impacts on children’s qual- ity of life (QOL), particularly related to psychosocial 1 Division of Otolaryngology–Head and Neck Surgery, University of functioning and development of peer relationships (Guralnick Wisconsin, Madison, Wisconsin, WI, USA et al., 1996). Children with communication disorders have been 2 Department of Otolaryngology, Vanderbilt University, Nashville, TN, USA shown to engage in active conversations less often, have lower rates of positive social behaviors, and be less successful in Corresponding Author: receiving appropriate responses to their social attempts (Gur- Susan L. Thibeault, Division of Otolaryngology–Head and Neck Surgery, University of Wisconsin, 5103 WIMR 1111 Highland Ave, Madison, WI alnick et al., 1996). Clefting of the lip and palate are some of 53705, USA. the more common birth anomalies and the most common Email: [email protected] 2 The Cleft Palate-Craniofacial Journal XX(X) these children to obtain normal speech and resonance; how- Previous research on children with cleft lip/palate has been ever, rates reported for velopharyngeal insufficiency (VPI) focused on their QOL, their speech characteristics, or how have ranged from 10.3% (Mahoney et al., 2013) to 25.6% peers/adults perceive their speech, with very little to none look- (McCrary et al., 2020), with rates as high as 32% for 2- ing into the relationship between QOL and speech characteris- staged repairs (McCrary et al., 2020). tics. The purpose of this study was to investigate the Velopharyngeal insufficiency typically causes hypernasal- relationship between QOL in children aged 3 to 18 with history ity, which negatively impacts social interaction, even in chil- of repaired unilateral/bilateral cleft lip/palate, cleft palate only, dren with mild hypernasality (Watterson et al., 2013). Research cleft of the soft palate only, and submucous cleft palate and has shown that children with a history of cleft lip/palate are perceptual speech characteristics. To measure QOL we utilized more likely to be teased by their peers, demonstrating further the VELO QOL instrument. This is a 23-item VPI-specific the potential psychosocial consequences of anxiety, self- QOL instrument with 6 domains, including speech limitations, esteem, depression, and behavioral problems (Hunt et al., swallowing problems, situational difficulty, emotional impact, 2007). Barr et al. (2007) concluded in their study of QOL in perception by others, and caregiver impact (Skirko et al., children with VPI that those with VPI and their parents 2012). This study was designed to investigate whether QOL reported a greater degree of impact to QOL as compared to of the participants, as measured by parent proxy reported (and age-matched peers without VPI. Bruneel and colleagues (2019) self-reported, if over 8 years) VELO scores, correlate with found that more negative speech outcomes were associated quantitative perceptual analysis of speech and if there is a with more negatively impacted QOL in children with VPI. correlation between certain features of speech (including nasal QOL has been found to improve following speech surgery in air emission, facial grimace, nasality/resonance, phonation/ patients with VPI (Bhuskute et al., 2017). Both patient and voice, and articulation) as measured by the Pittsburgh parent-reported health related QOL scores were found to be Weighted Speech Scales (PWSS). Our hypothesis was that worse in children with moderately to severely impacted speech children with more severely affected speech would have lower (Bickham et al., 2017). Taken together, speech production has QOL measures. an effect on QOL in children with cleft palate. Although research connecting perceived speech character- istics (ie, nasal air emissions, nasality, and articulation) and Methods QOL in children with cleft lip/palate is sparse, research specific to the impact of voice disorders on QOL has shown that chil- Study Design dren with voice disorders (ie, vocal fold nodules, vocal fold After obtaining approval from the institutional review board, paralysis, or paradoxical vocal fold motion) demonstrate a sta- we began prospective enrollment of participants aged 3 to tistically significant negative impact in their QOL as reported 18 years with history of a repaired palate (hard, soft, or sub- by both children and parents (Merati et al., 2008). Use of parent mucous), repaired at least 6 months prior to consent, who were proxy reports has been shown to be necessary for young chil- identified during their standard of care appointment with the dren as parents are thought to have a better understanding of Cleft and Craniofacial Anomalies Clinic. Informed consent their child’s problem, and parental concern is often the primary was obtained from parents or legal guardians and children older factor for decision-making regarding treatment (Boseley et al., than the age of 15 years, with assent obtained from children 2006; Watterson et al., 2013). Parent proxy reports have also aged 8 to 14 years. Parents acted as proxy and completed the been shown to reliably capture the QOL impact on children VELO about their child’s condition and QOL regardless of (Eckstein et al., 2011). In a study, on oral health-related QOL in their child’s age. Children older than the age of 8 years com- Ethiopian children born with orofacial clefts and their parents, pleted the VELO on their own in addition to the parent proxy. no significant difference was found in overall scores obtained Participation was voluntary and no compensation was provided from children and those obtained by parent proxy (Abebe et al., for participation. Exclusion criteria included children and/or 2018). Additionally, in a study designed to determine whether parents who were non-English speaking, children with a con- QOL is altered in children between the ages of 5 and 17 years firmed syndrome diagnosis, children with severe intellectual as measured by the Velopharyngeal Insufficiency Effects on disability, and/or children with a reading delay that would place Life Outcomes questionnaire (VELO) questionnaire as well as him or her below a third grade reading level confirmed by assess parent proxy and child agreement, Barr et al. (2007) parent/guardian. found no significant difference between parents and children’s judgment of the effects of speech limitations to QOL. They did, however, find that parent and child perceptions had some Data Collection degree of variance in that parents reported a lesser degree of Data points of interest included participant demographics (ie, impact of VPI on swallowing and an elevated degree of emo- age, gender, diagnosis, age at initial repair, revision surgical tional impact on their child’s QOL as compared to child self- history), VELO subscores and total scores for parents and chil- reports (Barr et al., 2007). Conversely, Bickham and colleagues dren older than 8 years, and perception of various speech char- found good agreement between parent and child reports of acteristics, as determined by a certified speech-language health-related QOL (Bickham et al., 2017). pathologist (SLP) using the PWSS. Demographic information Pedersen et al 3 was collected via review of medical records included the Table 1. Demographics of Study Population. child’s age at time of consent and gender. The type of cleft Age was also obtained and later simplified into 5 groups (unilateral Mean (SD) 9.5 (4.2) cleft lip/palate, bilateral cleft lip/palate, cleft palate only, cleft Sex of the soft palate only, or submucous cleft palate). Male 26 (45.9%) Female 22 (54.2%) Velopharyngeal insufficiency effects of life outcome. The VELO is a Diagnosis 23-item survey for children ages 8þ years and a 26-item survey Unilateral cleft lip/palate 27 (56.3%) for parents or guardians. The survey uses a 5-point Likert scale Bilateral cleft lip/palate 8 (16.7%) Cleft palate only 5 (10.4%) ranging from 0 (never) to 4 (almost always) to assess the degree Cleft of the soft palate only 5 (10.4%) to which each of the 5 subscales are problematic to the child with Submucous cleft palate 3 (6.3%) a history of VPI. The 5 subscales include speech limitations, Initial repair swallow problems, situational difficulty, emotional impact, and Mean age in months (SD) 18.7 (22.9) perception by others. Parent proxy questionnaires contained an Revision surgeries additional subscale assessing caregiver impact. Scores are cal- Mean (SD) 0.8 (0.9) culated by subtracting the sum of from 100 to receive a score that Abbreviation: SD, standard deviation. ranged from 0 to 100, with 100 representing better QOL (Skirko et al., 2012; Skirko et al., 2013; Skirko et al., 2015). velopharyngeal incompetence (Dudas et al., 2006). Twenty Speech assessment. Audio/visual recordings were a component percent of samples were chosen for repeated PWSS analysis of our cleft palate/craniofacial team standard of care speech for intrarater reliability. and resonance evaluation for all participants. These recordings contained full images of each child’s face. Recordings included Data Analysis syllable repetition, phrase repetition, counting, and conversa- tional speech sample, allowing for a comprehensive assessment The relationship between VELO total and PWSS total and sub- of nasal and non-nasal phonemes and changes in resonance scale scores was evaluated with Pearson correlation coeffi- across various levels of speech production. Younger partici- cients. We used analysis of covariance to evaluate whether pants repeated the phrases, while older participants read them. differences in VELO scores between parents and children were Participants were asked about their favorite movie, book, video related to cleft type (unilateral cleft lip/palate, bilateral cleft lip/ game, or activity. Attempts were made not to interpret sponta- palate, cleft palate only, cleft of the soft palate, and submucous neous speech. Although spontaneous speech samples were cleft palate), age, and gender. Parent and child VELO responses shorter for some of the younger participants, all recordings were compared using paired t tests to determine whether there included at least 15 syllables of spontaneous speech. Specific were differences between their responses. Intrarater reliability speech stimuli used are included in Online Appendix A. Videos for PWSS ratings was determined with Pearson correlation coef- were randomized and all direct portions of the video where ficient. All analyses were performed using SAS statistical soft- identifiers were present were removed. A licensed SLP (with ware version 9.4 (SAS Institute Inc). P values less than .05 were 9 years of experience working on a cleft palate/craniofacial considered to be statistically significant. team) performed the PWSS perceptual speech analysis. The rating was performed on the recording as a whole and full faces were not covered as facial grimace is an attribute that is Results assessed by the PWSS. The PWSS was selected due to its nature of systematically quantifying perceptual speech charac- Participant Demographics teristics (Dudas et al., 2006). This tool entails rating 5 speech Fifty-five participants were consented. Seven participants were attributes—nasal air emissions, facial grimace, nasality/reso- excluded based on incomplete questionnaires or poor video nance, phonation/voice, and articulation—which are then given quality. Final enrollment included 48 participants (26 males and relative weight toward a composite speech score. Composite 22 females) with a mean age of 9.5 (+4.02 years) and their scores are intended to be predictive of the competence of the parents. Participants were classified as 8 (16.67%) bilateral cleft velopharyngeal mechanism (Dudas et al., 2006). The PWSS lip/palate, 27 (56.25%) unilateral cleft lip/palate, 5 (10.42%) has been used to evaluate speech outcomes after secondary cleft of the soft palate only, 5 (10.42%) cleft palate only, and palate surgery (Noorchashm et al., 2006; Nayar et al., 2014; 3 (6.25%) submucous cleft palate. The mean age of primary Georgievska-Jancheska et al., 2016), after maxillary advance- surgery for those with bilateral cleft lip/palate, unilateral cleft ment (Chung et al., 2019), and to evaluate speech in children lip/palate, cleft of the soft palate only, and cleft palate only was with craniosynostosis (Naran et al., 2017). Scores are classified 9.22 + 4.04 months and 7 + 2.65 years (84 + 31.75 months) based on a range from 0 to >7 where 0 indicates velopharyngeal for those with submucous cleft palate. Lastly, there was an aver- competence, 1 to 2 equates to borderline competence, 3 to 6 age of 0.75 (+0.863) revision surgeries for these participants. equates to borderline incompetence, and >7 indicates Demographic information is summarized in Table 1. 4 The Cleft Palate-Craniofacial Journal XX(X)

Table 2. Comparison of Child and Parent VELO Scores.

VELO domain Child scores, mean (+SD), range Parent score, mean (+SD), range P value Speech limitations 75.53 (18.28), 25-100 75.79 (16.94), 4-100 .9529 Swallow problems 95.37 (7.75), 67-100 96.60 (9.63), 0-100 .4246 Situational difficulty 81.67 (15.32), 50-100 75.00 (20.10), 15-100 .1086 Emotional impact 94.91 (8.67), 69-100 90.74 (13.47), 25-100 .1665 Perception by others 95.37 (6.62), 81-100 93.75 (9.91), 44-100 .2943 Caregiver impact 87.50 (14.38), 50-100 Total score 86.27 (8.95), 68-99 81.81 (15.20), 20-100 .5216

Abbreviation: SD, standard deviation.

Velopharyngeal Insufficiency Effects of Life Outcome A 10 Mean, standard deviation (SD), and range of child and parent 9 scores for the VELO are provided in Table 2. Mean total scores 8 7 were 86.27 (+8.95) for child and 81.81 (+15.20) for parents. 6 All scores were within the 95% CI reported by Skirko et al. 5 (2012; 2013; 2015). No significant differences were measured 4 between parent and child on total or subscores. Lastly, there PWSS CHILD 3 was no significant difference between parent and children 2 r= -0.511, p = 0.0002 1 VELO scores related to cleft type, age, and gender (P ¼ .8873). 0 0 10 20 30 40 50 60 70 80 90 100

VELO TOTAL Pittsburgh Weighted Speech Scale B 6 Total PWSS mean and SD scores ranged from 1.38 to 1.96 with 5 highest mean response being reported for the subscore of nasal air emissions (0.52 + 0.95) followed by nasality/resonance 4 (0.42 + 0.85). Pittsburgh Weighted Speech Scale mean score 3 for articulation was 0.33 (+1.40), phonation/voice was 0.10

(+0.31) and nasality for 0. Articulation features that were CHILD PWSS 2 assessed and determined to be in error included reduced 1 r= 0.197, p = 0.325 intraoral pressure for plosives, production of glottal stops, pro- 0 duction of lingual palatal sibilants, omission of fricatives, and 0 102030405060708090100 production of pharyngeal fricatives, plosives, backing, snorts, VELO TOTAL inhalations, or exhalation substitutions. Overall, 25 (52%) participants received a total score of 0 Figure 1. Scatter plot showing correlation between parents’ scores indicating velopharyngeal competence, 13 (27%) received a (A) and child scores (B) for Total Pittsburgh Weighted Speech Scale score of 1 to 2 indicating borderline velopharyngeal compe- (PWSS) and Velopharyngeal Insufficiency Effects on Life Outcomes tence, 9 (19%) received a score of 3 to 6 indicating borderline questionnaire (VELO). Dotted line indicates best fit trend line. Pear- velopharyngeal incompetence, and 1 (2%) received a score >7 son coefficient ¼ r and P value provided within panel. indicating velopharyngeal incompetence. Intrarater reliability for phonation/voice and articulation was perfect (r ¼ 1). Excel- Table 3). Parent VELO total scores and PWSS articulation lent reliability for nasal air emissions (r ¼ 0.95) was deter- were significantly negatively correlated (r ¼0.37420, P ¼ mined with good reliability for nasality/resonance (r ¼ 0.84) .0088). No significant correlations between PWSS total and and PWSS total scores (r ¼ 0.82). child VELO total score were measured (Figure 1B). Further, no significant correlations were measured between PWSS total and VELO subscales or VELO total and PWSS subscales Pittsburgh Weighted Speech Scale and VELO (Table 3). To determine whether a relationship between perceptual fea- tures of speech and QOL exists, Pearson correlation coeffi- cients were calculated. A significant moderate negative Discussion correlation between PWSS total and parent VELO total scores This study was designed to investigate the relationship between (r ¼0.51103, P ¼ .0002) was measured (Figure 1A). The QOL in children aged 3 to 18 years with history of repaired VELO subscore comparisons for PWSS total score elicited cleft palate, as measured by self-report (children 8þ years) and significant correlations for all 6 categories (mild to moderate; parent proxy, and perceptual speech characteristics as Pedersen et al 5

Table 3. Relationship Between PWSS and VELO. a main predictor of increased behavioral problems, decreased happiness, and increased anxiety. In Ireland, Lee et al. (2017) Correlation found that children with cleft palate with reduced speech intel- Comparisons coefficient P value ligibility were perceived by their peers as less healthy, not as PWSS and Parent VELO good looking, and probably had fewer friends. Lastly, the World PWSS Total & VELO Speech Limitations 0.470 .0008a a Health Organization (2001) stated that peer attitudes are one of PWSS Total & VELO Swallow Problems 0.544 <.0001 the most important environmental factors that will either facil- PWSS Total & VELO Situational Difficulty 0.358 .0124a PWSS Total & VELO Emotional Impact 0.016 .0162a itate or hinder overall social functioning and participation of PWSS Total & VELO Perception of Others 0.470 .0008a individuals with speech problems. Our data demonstrate that PWSS Total & VELO Caregiver Impact 0.370 .0096a there is a relationship between speech characteristics and QOL VELO Total & PWSS Nasal Air Emissions 0.275 .0586 and in combination with other research, suggests these children VELO Total & PWSS Nasality/Resonance 0.274 .0589 with cleft palate may be at risk for negative psychosocial impact VELO Total & PWSS Phonation/Voice 0.046 .7554 a (Starr et al., 1984; Hunt et al., 2007; Watterson et al., 2013). VELO Total & PWSS Articulation 0.374 .0088 We did not find a relationship between perceptual speech PWSS and Child VELO PWSS Total & VELO Speech Limitations 0.259 .1918 analysis and child self-reported QOL. Overall, our child- PWSS Total & VELO Swallow Problems 0.219 .2726 reported QOL scores were higher than parent-proxy reported PWSS Total & VELO Situational Difficulty 0.046 .8196 QOL scores, albeit these differences were not significant. One PWSS Total & VELO Emotional Impact 0.136 .4957 reason for the lack of significance may be the population PWSS Total & VELO Perception of Others 0.206 .3021 sampled. Many of the children in the study were perceptually VELO Total & PWSS Nasal Air Emissions 0.173 .3889 rated to be perceived as having velopharyngeal competence VELO Total & PWSS Nasality/Resonance 0.014 .9429 (52%) or borderline velopharyngeal competence (27%). How- VELO Total & PWSS Phonation/Voice 0.215 .2827 VELO Total & PWSS Articulation 0.088 0.4541 ever, it has shown that even mild hypernasality can be associ- ated with negative social factors such as being perceived to be a Abbreviation: PWSS, Pittsburgh Weighted Speech Scale. good partner, fit in with friends, get good grades, be teased, and a statistically significant p<0.05. have an easy time making friends (Watterson et al., 2013). The lack of relationship found could also be due to the sample size measured by the PWSS. Overall, we found negative correla- as there were 48 parent VELO questionnaires completed, but tions between perceptual speech analysis and QOL by parent only 27 children met the age criteria for the youth question- naire. Further, some of the QOL findings relate to overall proxy, but not from the child’s perspective. Further, this neg- self-image, and not just speech. Typically, preadolescents and ative relationship measured in parents appears to be driven by adolescents are developing their self-image (Levine et al., articulation versus other speech measures as there was a sig- 2002). There have been conflicting findings on satisfaction nificant negative correlation between articulation scores on the with facial and body image in children and adolescents with PWSS and QOL by parent proxy. These findings suggest that cleft lip/palate. For example, Leonard et al. (1991) found that as a child’s articulation is scored closer to normal, parents overall, children, and adolescents with cleft lip/palate had aver- report improved QOL in their children specifically for speech age to above average self-image, but adolescent females had limitations, swallow problems, and perception of others. more negative self-image in comparison with younger children Our findings are supported by others’ work. Most recently in and adolescent boys. Adolescents with cleft lip/palate were Belgium, moderate to strong negative correlations were found found to have an average or above average self-image (Gussy between total VELO parent scores and speech variables; as and Kilpatrick, 2006). Conversely, adolescents with cleft lip/ speech outcomes were judged to be poor, QOL decreased (Bru- palate had significantly lower scores on a measure of psycho- neel et al., 2019). More specifically, this recent study reported social impact of dental aesthetics than peers without a cleft lip significant correlations between total VELO parent scores and or palate also requiring orthodontic treatment (Raghavan et al., speech understandability, passive cleft speech characteristics, 2019), but similar life satisfaction scores. Children and adoles- speech acceptability, and the need for speech therapy (Bruneel cents with cleft lip/palate have been found to be dissatisfied et al., 2019). The relationship between QOL and speech/articu- with their appearance, reported worse social adjustment, and lation is of interest as other investigations have determined that lower self-esteem than peers (Richman, 1983; Broder et al. there is a relationship between speech intelligibility and subse- 2007; Kapp-Simon, 1995). These findings seem to suggest that quent perception by others. Bickham et al. (2017) report that while children and adolescents with cleft lip/palate may have patients who receive worse scores on speech analysis have average to above average self-image, there are specific aspects more anger, depression, and difficulty with peer relationships. of self-image or self-perception that present more challenges. Parent and child reports of QOL have shown improvements These are difficult to parse out using many of the instruments following surgery to correct speech, suggesting that improve- available. ment in speech has a positive impact on QOL (Bhuskute et al., We did not find differences between parent and child’s QOL 2017). Hunt et al. (2007) determined that in children with cleft scores and our findings are similar to Abebe et al. (2018), who lip/palate, being teased because of impaired speech quality was found no significant difference between parent and child 6 The Cleft Palate-Craniofacial Journal XX(X) questionnaires; however, our results differ from Barr et al. perceptual analysis of speech improves as measured by the (2007) who found significant differences between parent and PWSS, QOL improves moderately as judged by parents. There child reports for swallowing problems, with children perceiv- was no relationship between these 2 factors in children between ing a greater degree of impact. Our largest nonsignificant mean the ages of 8 and 18 years. Further research that includes larger differences were for the subscale situational difficulty. Situa- sample sizes and diversity in severity are needed to further tional difficulty assesses if a child is understood in various elucidate the relationship between perceptual analysis of situations/contexts such as on the phone or in a car. Differences speech and participant perception of QOL. found between parent and child report may be due to differ- ences in their understanding of the problem, its treatment, and Acknowledgments its degree of impact as children have less experience of health, The authors gratefully acknowledge Glen Leverson for his expert illness, and medical care (Eiser, 1990; Eiser, 1997). Parents are assistance with statistical analysis for this study. able to make reasonably accurate judgments in terms of the family and sibling relationships; whereas, the child can make Declaration of Conflicting Interests judgments regarding their experience with symptoms, their The author(s) declared no potential conflicts of interest with respect to relationships with their peers, and future concerns (Eiser, the research, authorship, and/or publication of this article. 1990; Eiser, 1997; Hunt et al., 2007; Abebe et al., 2018). One unexpected finding in this investigation was the degree Funding to which PWSS total was negatively correlated with the parent The author(s) disclosed receipt of the following financial support for VELO subcategory of swallow problems but not the children’s the research, authorship, and/or publication of this article: This work reporting. This finding is inconsistent with the overall findings was supported by the Diane M. Bless Endowed Chair in Otolarynol- of velopharyngeal competence in our participant population ogy Head and Neck Surgery. which would infer normal swallowing and with overall high VELO scores. It is possible that even a few instances of food ORCID iD coming out of the nose could be extremely salient and influence Susan L. Thibeault, MS, PhD, CCC-SLP https://orcid.org/0000- the VELO score unduly for the parents versus children. Unfor- 0002-9046-4356 tunately, this study did not specifically evaluate eating or swal- lowing function, it is not possible to correlate this aspect of Supplemental Material QOL with any objective measures; further investigation is Supplemental material for this article is available online. warranted. Limitations of this study include video recording content References inconsistency based on age, as younger children had more Abebe ME, Deressa W, Oladugba V, Owais A, Hailu T, Abate F. Oral difficulty following protocol and were often reluctant to speak health-related quality of life of children born with orofacial clefts in front of a camera. Any videos that did not include a con- in Ethiopia and their parents. Cleft Palate Craniofac J. 2018;55(8): versational speech sample of at least 15 syllables of sponta- 1153-1157. neous speech were excluded. All videos analyzed in this study Barr L, Thibeault SL, Muntz H, de Serres L. 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