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Five-year speech and language outcomes in children with cleft lip-palate
Accepted Manuscript Five-Year Speech and Language Outcomes in Children with Cleft Lip-Palate Benjamas Prathanee, PhD., Tawitree Pumnum, BSc., Cholada Seepuaham, BSc., Pechcharat Jaiyong, BSc. PII:
S1010-5182(16)30172-X
DOI:
10.1016/j.jcms.2016.08.004
Reference:
YJCMS 2456
To appear in:
Journal of Cranio-Maxillo-Facial Surgery
Received Date: 7 April 2016 Revised Date:
30 June 2016
Accepted Date: 3 August 2016
Please cite this article as: Prathanee B, Pumnum T, Seepuaham C, Jaiyong P, Five-Year Speech and Language Outcomes in Children with Cleft Lip-Palate, Journal of Cranio-Maxillofacial Surgery (2016), doi: 10.1016/j.jcms.2016.08.004. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Five-Year Speech and Language Outcomes in Children with Cleft Lip-Palate
Benjamas Prathanee PhD.*,
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Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand, e-mail address: [email protected] Tawitree Pumnum, BSc.
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Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
Cholada Seepuaham, BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
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Pechcharat Jaiyong, BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
*Corresponding author: Benjamas Prathanee, Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand, 40002, Tel. 66-43-363565, Fax: 66-43-202490, e-mail address: [email protected]
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Preschool Speech Outcomes in Children with Cleft Lip-Palate Benjamas Prathanee PhD.,
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Tawitree Pumnum BSc., Cholada Seepuaham BSc., Pechcharat Jaiyong BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University,
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Khon Kaen, Thailand
1. Introduction
The worldwide incidence of cleft lip/palate was found to be between 0.11 and 2.65/1,000 live births (Hobbs et al., 2001, Chowchuen and Godfrey, 2003, Natsume and Tolarova 2006).
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Cleft lip and palate (CLP) is indeed a major public health concern in Thailand, where the incidence of cleft lip/palate is between 1.10 and 2.49/1,000 live births (Chuangsuwanich et al., 1998). CLP has several impacts on physical and psychological skills including nasolabial
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appearance, bone structure, swallowing, speech and language, hearing, dental, occlusion, delayed development, social growth, quality of life, as well as affecting individual and country
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economics. Interdisciplinary approaches from birth to 19 years of age are needed as a complete remedy.
Three important main outcomes of treatment for CLP are normal configuration, normal
speech functions and good quality of life. Surgery is needed for lip correction at age 3 months and palatoplasty at 1 year of age for relief of the stigmas of facial abnormality and to expect normal function of speech production. Residual speech and language problems after surgery,
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however, are of critical concern. The rates of speech and language abnormalities including delayed speech and language development were in the range of 16-19 %, articulation defects 2390% (Lohmander-Agerskov and Soderpalm, 1993, Schonweiler et al., 1999, Prathanee et al.,
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2013), resonance disorders 27-43 % (Schonweiler et al., 1999, Prathanee et al., 2013, Prathanee et al., 2014) and voice disorders 0.6-50 % ( Lohmander-Agerskov et al., 1995, Hamming et al., 2009, Robison and Otteson, 2011, Prathanee et al., 2013, Prathanee et al., 2014). Compensatory
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articulation disorders (CAD) are generally the most common problems for children with clefts.
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Children with CAD also presented with significant language delays (Pamplona et al., 2000). Children aged 4-8 years are in the transition phase from the pre-school to early school periods in which the development of cognitive skills and critical adaptation to others for living in society occurs (Child and Adolescent Health Service, 2013). Speech and language skills are crucial factors that affect the quality and appropriateness of character in school during this
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period. Based on Khon Kaen University Center’s protocol that is concerned with the balance of these outcomes, the schedules for labioplasty is at 3 months of age, palatoplasty and veloplasty at around 12 months of age are conducted to improve configuration, speech, language, hearing
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abilities and quality of life as early as possible. The investigation of speech and language
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outcomes at 5 years is necessary for further management and remedy planning to support adaptation in school, and feedback for reviewing the treatment protocols to improve clinical outcomes.
The aim of the present study was to investigate speech and language outcomes for
children with CLP aged around 5 years or who were in the transition phase from the pre-school to early school periods.
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2. Materials and methods After the research protocols were approved (August 7, 2009) by the Human Research
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Ethics Committee of Khon Kaen University in accordance with the Helsinki Declaration (HE521052), informed consents were obtained from the subjects’ legal representatives, and 38 children with CLP aged 4-7 years 8 months were recruited for this study. Children’s speech and
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language outcomes were assessed by the principal investigator and a qualified speech and
language pathologist (SLP) who was a research assistant and who had clinical experience related
then followed as shown below:
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to speech and language in cleft lip and palate children for more than 20 years. The protocol was
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Oral examination and facial grimaces
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Speech and language abilities with perceptual assessment of speech for clefts using
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the Thai Universal Parameters of Speech Outcomes for People with Cleft Palate and outcomes were recorded in case record forms that were developed for the standard test (Henningsson et al., 2008, Prathanee et al., 2011) included:
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Articulation: 2 rating scales. Each sound was evaluated by perceptual assessment. Score
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0=normal articulation; 1= abnormal articulation in any type of standard patterns for cleft palate speech (Henningsson, et al., 2008, Prathanee B et al., 2011). Resonance: 5 rating scales. Score 0=Normal: there was no perceptual evidence of cleft
type speech and does not exceed nasality heard in regional speech; 1 = Mild: Nasality exceeds regional speech nasality; 2 = moderate: hypernasality is perceived as pervasive and draws attention to itself and away from the message; 3 = severe: hypernasality perceived as pervasive and interferes with speech understandability.
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Voice: 2 scales. Voice assessment was performed by using the perceptual assessment or GIRBAS (Grade = overall impression, I: Instability or fluctuation of voice, R: Roughness, hoarseness, B: Breathiness, breathy voice, A: Asthenia or a weak voice or speaking with minimal
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air volume, S: Strain or forced or stressed voice). Each parameter was scored as 0= normal; 1=mild; 2=moderate; 3=severe. GIRBAS is a popular and reliable perceptual scale (Webb et al., 2004). Total score of GIRBAS: 0: normal voice; 1-18: abnormal voice. Score 0= normal; 1=
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abnormal voice).
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Intelligibility: 4 rating scale (0= normal: intelligibility or the whole speech sample was always easy to understand; 1= mild or the whole speech sample was occasionally hard to understand; 2= moderate or the whole speech sample was often hard to understand; 3= severe or the whole speech sample was hard to understand most or all of the time.
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Nasal emission/turbulence: 3 rating scales. Score 0=none; 1= intermittent or variable (occasionally heard on high pressure consonants and an error production needs to occur more than once for it to be documented of reliable error type); 2 = frequent or pervasive (heard on
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production of many high pressure consonants).
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- Language screening test (adapted Thai Early Language Milestone) (Lorwatanapongsa et al., 2011) and UTAH Test (Mecham and Jones, 1967)].
Statistical Analysis
The main outcomes were the numbers of children with CLP who had language delay, abnormal understandability, resonance abnormality, voice disturbance, and articulation defects.
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The oral examination described characteristics of oral configurations. Perceptual assessments were also scored as Resonance normal (0), abnormality if resonance was scored ≥1 hyponasality (-1), mild hypernasality (+1), moderate hypernasality (+2), severe hypernasality (+3) ; Voice as
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normal (0) and abnormal if any parameter of the voice was scored ≥ 1 (1: Understandability as normal (0), abnormality if understandability was scored ≥ 1 (1) (mild or occasionally hard to understand = 1, often hard to understand = 2, hard to understand most or all of the time = 3);
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Articulation defects as normal articulation development (0), at least 1 articulation error (1).
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Double entries and incorrect filing of data were corrected. Data analyses were performed by using numbers and percentages and analyzed by a Strata program.
3. Results
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Participants aged 4-8 years who were registered in the Khon Kaen University Center for Cleft Lip-Palate and Craniofacial Anomalies were enrolled in this study. Thirty-eight children with CLP were investigated for speech and language outcomes. General characteristics of
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children with CLP are displayed in Table 1.
Table 1
Children with CLP were 20 females and 18 males. Twenty-one children (55.26%) were
left CLP; 6 (15.79 %) were right CLP; and 11 (28.95 %) were bilateral CLP. With the exception of C 39 (Treacher Collins Syndrome) and C 40 (Facial cleft), seven of 36 children (19.44 %) presented with oronasal fistulas, two of them (5.26 %) had tongue-ties and 23 of 36 children
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(63.38 %) had malocclusions Class III. With the exception of the complicated or syndromic cases, C39 and C 40, data were
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described and analyzed based on total of 36 children. Five-year speech and language outcomes
nasometer and are displayed in Table 3.
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Table 2
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in children with CLP were assessed and are presented in Table 2. Resonance outcomes were by
Table 2 shows that 3 children had delayed speech and language development, 18 had an abnormality of understandability, 13 had resonance abnormalities or hypernasality, 11 had voice abnormality, and 34 had articulation defects. Thirty five of 36 children with CLP (97.22 %), with
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the exception of C28, incorrectly produced postalveolar trill or rolling R (/r/). The prevalence of articulation defects (34 of 36 children) with exception of /r/ was 94.44 %. C23 had only an error
Table 3
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of /r/.
There were 14 of 36 children with CLP (38.89 %) who had nasal emission or nasal
turbulence. Eight of 14 children (57.14 %) had hypernasality on perceptual assessment with nasal air emission or turbulence. Interestingly, 6 of 36 children (42.87 %) had normal resonance with some air emission or turbulence. Regarding speech and language disorders, prevalence rates are presented in Tables 4 and
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5. Table 4
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Articulation errors were the most common speech defects in children with CLP, and the average number of articulation errors among children with CLP was 12.69 % or a median 12.25 (Min= 0: Max =33), followed by the abnormality of understandability, hypernasality, and voice
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disorders.
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The prevalence of speech defects based on severity is shown in Table 5.
Table 5
Table 5 shows that the most common degree of abnormality was mild both in
4. Discussion
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these children.
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understandability and resonance disorders. Voice abnormality had the higher prevalence among
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Primary surgical protocol evaluation should be done at a patient age of approximately 5
years to reduce biases such as secondary surgeries or orthodontic treatment, and results are therefore obtained with minimal negative effects on facial growth (Dissaux, 2016). Optimal speech outcome is one of the most important functional goals of primary cleft surgery. Assessments of speech outcomes used in previous studies (Persson et al., 2002, Nyberg et al., 2010, Klinto et al., 2011, Britton, Albery et al., 2014, Dissaux, Grollemund et al., 2016)
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were used for estimation the main outcomes of this study. Data of 36 children with CLP, without heterogeneous cases (C39 and C40), are presented for assessment to plan for future appropriate treatment. The fistula rate was 19.44 % and was in the low- to mid-range compared to previous
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studies, range 2.4 % to 60% (Murthy et al., 2009, Stewart et al., 2009, Eberlinc and Kozelj, 2012, Hortis-Dzierzbicka et al., 2012). Besides the surgical technique, there might be other factors that affect the oronasal fistula rate including type and severity of clefts and experience of surgical
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skills (Murthy et al. 2009). These factors should be considered for surgical planning.
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For main outcomes, 3 children (7.89 %) had delayed speech and language development. This prevalence was low when compared to previous studies (range 13 -. 92 %) (Schonweiler et al., 1996, Vallino, Zuker et al., 2008, Ruiter et al., 2009, Rullo et al., 2009, Young et al., 2010, Prathanee et al., 2013). The early diagnosis and intervention program was effective for language stimulation and supports a previous finding (Ruiter, et al. 2009). Eighteen children with CLP (50
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%) had unclear speech or an understandability deviation. This was in the range of previous reports 44-63 % (Schuster et al., 2006, Normastura et al., 2008, Rullo et al., 2009). For resonance disorders, 14 of 38 children (36.11 %) had hypernasality and this was also in the range of
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previous reports of 20-43.30 % (Grunwell et al., 2000, Kummer, 2001, Sell et al., 2001,
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Prathanee, 2002, Kummer, 2008, Phua and de Chalain, 2008, Prathanee, 2012). This might validate that the intravelar veloplasty technique that is used in the KKU center was an effective surgical technique and seems to have less of a negative impact on maxillary growth and give good speech outcomes (Dissaux et al., 2016). In all children with CLP’s nasalance scores, Laying Hen (a load of every sound in Thai language) and My House (the load of both oral and nasal sounds), were out of normal range (more than mean ± 1SD) that agreed with perceptual assessment outcomes (mild and moderate hypernasality). This indicated that nasalance scores
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and perceptual assessments might be representative of each other. Overall, 11 of a total 36 children (30.56 %) had voice abnormalities and this was at a
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higher rate than previous studies, which ranged between 5.5-20.8 % (Hocevar-Boltezar et al., 2006, Hamming et al., 2009, Robison and Otteson, 2011, Prathanee B et al., 2013). Seven of 11 children (63.64%) with CLP had voice abnormalities with hypernasality (Table 2), however, the
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rest of them (4 of 11 or 36.36 %) had voice disorders and normal resonance by perceptual
assessment. This supported the theory that patients with CLP are at risk for voice disorders from
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both laryngeal hyperfunctions as a compensatory articulation mechanism for VPI ( Leder and Lerman, 1985, D'Antonio et al., 1988) and multifactorial variables related to voice abnormality. This occurs in clefts and includes gastroesophageal reflux diseases (Karkos et al., 2007, Fisichella, 2015) and might not relate to velopharyngeal insufficiency (Hamming et al., 2009).
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This needs to be confirmed by further study.
Cases of children with CLP who had a high rate of articulation errors with hypernasality of ≥ 12 articulation defects were found in 8 of 20 children (40.00 %), and the rest of them (12
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children or 60 %) had a high rate of articulation errors with normal resonance. This revealed that articulatory defects in CLP were not only caused by VPI, but might also be related to
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mislearning from incomplete kinesthetic patterns. For the other two patients, C39 and C40 (Treacher Collin Syndrome and Facial Cleft),
these patients had the largest number of articulation errors (21 and 18 sound errors) (Table 2), resulting in abnormal intelligibility. Even with palatoplasty for correction of velopharyngeal anatomy and good function that results in normal resonance (C39) and mild hypernasality (C40) without audible nasal emission or turbulence (Table 3), they might still have the limitation of
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oral structure from syndromic disease that is the cause of severe articulation errors and the need for long term speech therapy.
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There are limitations of speech services in Thailand, especially in the Northeast area composed of 21 cities and 21 million people, where the only tertiary level speech center and speech and language pathologists (SLP) are in the area where this study was done. There have
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been a few SLPs outside the KKU hospital center in the last 30 years. Therefore, most children with CLP received or no or little speech therapy, approximately one 30-minute session every 1-2
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months. The majority of them had not received continuous and long term speech therapy because 1) They could not afford expenses, e.g., living, transportation expenses, salary compensation etc., 2) It was a long distance to get speech services of 3-4 hours for a one way trip (Prathanee et al., 2006). Speech and language defects might be decreased if children with CLP could get appropriate speech therapy following the standard protocols. These data should provide feedback
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and stimulate more direct attention to the revision of protocols, including early speech and language intervention programs to solve compensatory articulation disorders on time and prevent long-term compensatory speech defects which need a longer and more difficult management.
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Successful speech camps or the Community-Based Speech Therapy Model are appropriate
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problem solving models (Pamplona et al., 1999, Pamplona et al., 2005, Prathanee et al., 2006, Prathanee et al., 2010, Prathanee et al., 2014). In addition, obligatory speech disorders caused from VPI that make incomplete kinesthetic patterns require early physical management and an early interventional speech program.
5. Conclusion
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Speech outcomes including articulation disorders, resonance disorders, voice abnormalities and understandability problems were of high prevalence. Early intervention programs and surgical techniques should be critically reviewed and the need further for
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prospective studies to improve speech outcomes for children with CLP should be pursued.
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Conflicts of interest:
Funding support:
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None
Cleft Lip/Palate and Craniofacial Abnormalities Center of Khon Kaen University
Acknowledgement
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Associate with “Tawanchai Foundation”.
The researchers thank the parents and children who participated in this project and Cleft
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Lip/Palate and Craniofacial Abnormalities Center of Khon Kaen University associated with “Tawanchai Foundation” for research and funding support. We would like to acknowledge Prof.
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James A. Will, for editing the MS via Publication Clinic KKU, Thailand.
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Thanaratsuthikul D: Artculation test. Master Degree, The Degree of Master of Art
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(Communication Disorders), Bangkok: Graduate School, Mahidol University. 1998. Vallino LD, Zuker R, Napoli JA: A study of speech, language, hearing, and dentition in children with cleft lip only. Cleft Palate Craniofac J 45 : 485 494, 2008.
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Webb AL, Carding PN, Deary IJ, MacKenzie K, Steen N, Wilson JA: The reliability of three perceptual evaluation scales for dysphonia. Eur Arch Otorhinolaryngol 261: 429-434, 2004. Young SE, Purcell AA, Ballard KJ : Expressive language skills in Chinese Singaporean
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456 464, 2010.
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preschoolers with nonsyndromic cleft lip and/or palate. Int J Pediatr Otorhinolaryngol 74 :
Table 1. Characteristics of children with clefts No.
Age
Gender
Diagnosis
Oral Examination
ACCEPTED MANUSCRIPT
(years & months)
- Lip contraction C 01
5; 10
Female
Lt.CLP
- Short uvula and active movement - Malocclusion Class III
Lt.CLP
C 04
4;5
Male
BCLP
C 05
5; 3
Male
Lt.CLP
C 06
5; 9
Female
Lt.CLP
- Bifid uvula and slight movement
Lt.CLP
- Short uvula and slight movement - Short uvula and active movement
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4
- Lip contraction
Female
SC
C 03
- Malocclusion Class III
- Bifid uvula and active movement - Malocclusion III
- Lip contraction
C 10
7; 8
Female
- Bifid uvula and active movement
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- Malocclusion III - Lip contraction
C 11
4; 3
Male
BCLP
- Oronasal fistula
- Bifid uvula and slight movement - Malocclusion III
C 20
C 21
6; 1
7; 1
Lt.CLP
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7; 1
Male
Male
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C 16
4; 4
AC C
C 12
Male
BCLP
- Lip contraction - Short uvula and active movement - Lip contraction and limited movement - Bifid uvula and slight movement - Malocclusion Class III - Lip contraction
Lt.CLP
- Bifid uvula and active movement - Malocclusion Class III - Lip contraction and limited movement
Female
BCLP
- Short uvula and active movement - Malocclusion Class II - Oronasal fistula
C 22
5; 10
Male
BCLP
C 23
5; 4
Female
Lt.CLP
- Lip contraction - Short uvula and slight movement - Lip contraction - Bifid uvula and slight movement
ACCEPTED MANUSCRIPT - Lip contraction C 24
6; 1
Female
Lt.CLP
- Bifid uvula and active movement - Oronasal fistula - Lip contraction
C 25
4; 6
Female
Lt.CLP and
- Short uvula and slight movement - Malocclusion Class II
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- Tongue tie
- Lip contraction C 26
5; 9
Male
Rt.CLP
- Short uvula and active movement - Malocclusion Class III
C 27
5; 9
Female
Lt.CLP
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- Lip contraction
- Oronasal fistula
- Bifid uvula and active movement
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- Alveolar Cleft
- Lip contraction
C 28
Male
5
Lt.CLP
- Short uvula and slight movement - Malocclusion Class III - Lip contraction
C 31
C 32
Lt.CLP
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Female
4
Female
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C 30
5; 3
5; 9
AC C
C 29
4; 7
Female
BCLP
- Short uvula and slight movement - Oronasal fistula - Lip contraction - Short uvula and active movement - Malocclusion: open bite & missing upper teeth - Lip node/bumpy
BCLP
- Oronasal fistula - Short uvula and active movement - Malocclusion Class III - Lip contraction and limited movement
Male
BCLP
- Short uvula and active movement - Malocclusion Class III - Tongue Tie
C 33
5; 8
Female
Rt.CLP
C 34
4; 8
Male
Lt.CLP
- Lip contraction - Short uvula and active movement - Short uvula and active movement - Malocclusion Class III
ACCEPTED MANUSCRIPT - Lip contraction C 35
Female
4; 11
Lt.CLP
- Short uvula and active movement - Malocclusion Class III - Lip node or bumpy
C 36
Female
4; 10
Rt.CLP
- Bifid uvula and active movement - Malocclusion Class III
C 39
5
Male
Female
4; 10
Lt.CLP
BCLP and Treacher Collins Syndrome
- Bifid uvula and active movement - Malocclusion Class III
- Lip contraction and limited movement - Oronasal fistula - Bifid uvula and slight movement
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C 38
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- Right Alveolar Cleft
- Malocclusion Class III
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- Missing upper teeth - Lip contraction
C 40
4; 4
Male
Lt.CLP and Facial cleft
- Bifid uvula and slight movement - Malocclusion - Left Alveolar Cleft
C 42
4; 9
4; 2
C 44
4; 3
C49
AC C
C 48
Male
4; 4
Lt.CLP
Rt.CLP
Male
BCLP
Female
Rt.CLP
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C 43
C 47
Female
4; 11
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C 41
- Short uvula and active movement
- Bifid uvula and active movement - Malocclusion Class III - Right Alveolar Cleft - Lip contraction - Malocclusion Class III - Short uvula and active movement - Lip contraction
Male
BCLP
- Short uvula and active movement - Malocclusion Class III
4; 8
Male
Rt.CLP
5; 3
Male
Lt.CLP
- Lip contraction - Bifid uvula and active movement - Short uvula and active movement - Malocclusion Class III - Lip contraction
C 51
5; 4
Female
Lt.CLP
- Oronasal fistula - Bifid uvula and active movement
ACCEPTED MANUSCRIPT - Malocclusion Class III - Lip contraction C 52
4; 1
Female
Lt.CLP
- Bifid uvula and slight movement - Malocclusion Class III
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Lt CLP: left cleft lip and palate; Rt. CLP: Right cleft lip and palate; BCLP: bilateral cleft lip and palate.
ACCEPTED MANUSCRIPT Table 2. Five-year speech and language outcomes in children with CLP No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors
Pass
Normal
Normal
C 03
Pass
Normal
Normal
C 04
Pass
Mild
Normal
Abnormal
17- 2-0-0=19
C 05
Delayed
Mild
Mild
Normal
16-2-0-0=18
C 06
Pass
Normal
Normal
Normal
8-2-4-0= 14
C 10
Pass
Mild
Moderate
Abnormal
17-2-0-0=19
C 11
Delayed
Mild
Moderate
Normal
13-3-1-0=17
C 12
Pass
Normal
Normal
Normal
9-2-0-0=11
C 16
Pass
Mild
Mild
Abnormal
12-2-0-0= 14
C 20
Pass
Normal
Normal
Normal
7-2-2-0=11
C 21
Delayed
Normal
Mild
Normal
7-0-1-0=8
Pass
Normal
Normal
Normal
16-1-0-0=17
C 23**
Pass
Normal
Moderate
Normal
1-0-0-0=1
C 24
Pass
Normal
Mild
Normal
8-3-0-0=11
C 25
Pass
Normal
Normal
Normal
10-0-0-0=10
C 22
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Normal
14-2-0-1=17
Normal
15-3-4-1 =23
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C 01
AC C
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I-F-V-T*
ACCEPTED MANUSCRIPT No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors
Pass
Mild
Normal
C 27
Pass
Mild
Normal
C 28
Pass
Normal
Normal
C 29
Pass
Moderate
Moderate
Abnormal
17-0-0-0=17
C 30
Pass
Mild
Normal
Normal
11-3-0-0=14
C 31
Pass
Mild
Normal
Abnormal
8-2-1-0=11
C 32
Pass
Severe
Normal
Abnormal
16-1-0-0=17
C 33
Pass
Mild
Normal
Normal
10-1-0-0= 11
C 34
Pass
Mild
Normal
Normal
11-1-0-0=12
C 35
Pass
Normal
Mild
Abnormal
6-2-0-0=8
C 36
Pass
Normal
Mild
Normal
7-2-0-0=9
C 38
Pass
Normal
Normal
Normal
7-2-1-0=10
Pass
Mild
Normal
Normal
15-2-4-0=21
Pass
Severe
Mild
Abnormal
17-1-0-0=18
C 41
Pass
Normal
Normal
Normal
8-1-0-0=8
C 42
Pass
Normal
Normal
Normal
2-1-1-0=4
C 39
C 40
Normal
Normal
7-2-0-0=9
Normal
0-0-0-0=0
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12 -2-0-1=15
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C 26
AC C
I-F-V-T*
ACCEPTED MANUSCRIPT No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors I-F-V-T*
Pass
Severe
Mild
Abnormal
C 44
Pass
Mild
Mild
C 47
Pass
Mild
Normal
C 48
Pass
Mild
Normal
Normal
11-2-1-0=14
C 49
Pass
Normal
Normal
Normal
11-2-1-0=14
C 51
Pass
Severe
Normal
Abnormal
11 -1-0 -0 =12
C 52
Pass
Normal
Mild
Abnormal
13-2-1-0 =16
18-5-7-3-0=33
Normal
17-2-2-0=21
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Abnormal
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C39: Treacher Collin Syndrome: 40: Facial cleft
* I: Initial sound; F: Final sound; V: Vowel; T: Tone
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**: Only articulation error was postalveolar trill or rolled R or rolling R (/r/)
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11-1-1-0=13
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C 43
Table 3. Perceptual resonance assessment, nasalance scores and audible nasal emission/turbulence
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Audible emission/turbulence& Word Sentence
My house
Winter
Laying hen
Resonance
C 01
54
59
23
Normal
1
1
C 03
22
35
8
Normal
0
1
C 04
43
53
24
Normal
1
1
C 05$
52
60
38
Mild
1
1
C 06
37
52
20
Normal
0
0
C 10*
54
56
47
Moderate
0
0
C 11*
66
68
41
Moderate
1
1
C 12
31
50
16
Normal
0
0
C 16$
57
57
46
Mild
2
2
C 20
29
46
16
Normal
0
0
C 21
58
66
45
Mild
1
1
C 22
36
46
17
Normal
0
0
C 23
52
55
46
Moderate
1
1
C 24
60
70
58
Mild
0
0
C 25
48
52
22
Normal
0
0
39
54
21
Normal
0
0
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EP
C 26
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Patient No.
41
58
17
Normal
0
0
C 28
39
56
31
Normal
0
0
C 29
40
41
35
Moderate
0
0
C 30
32
40
31
Normal
0
0
C 31
33
49
14
Normal
0
0
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C 27
ACCEPTED MANUSCRIPT
C 32
52
74
Normal
Resonance
0
0
Audible emission/turbulence& Word Sentence
My house
Winter
Laying hen
31
50
9
C 34
38
46
72
Normal
0
0
C 35
46
51
31
Mild
2
2
C 36
55
62
31
Mild
0
1
C 38
28
49
9
Normal
0
0
C 39
40
37
22
Normal
0
0
C 40
44
42
C 41
38
57
C 42
28
46
C 43
44
62
C 44
37
C 47
48
C 48
42
C 49
0
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SC
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Normal
0
36
Mild
0
0
18
Normal
0
0
14
Normal
1
1
51
Mild
0
0
53
44
Mild
0
0
54
36
Normal
0
0
54
30
Normal
1
2
Normal
0
0
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C 33
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Patient No.
64
N/A
N/A
N/A
36
46
24
Normal
0
1
C 52
40
47
23
Mild
0
1
Mean (SD.)
42.76 (10.76)
52.19 (8.06)
30.81 (16.36)
95 % Confident Interval
39.17- 46.34
49.50- 54.88
25.35-36.27
Norm{Prathanee, 2003 #123}
35.6 (5.9)
51.1 (6.4)
14.3 (5.8)
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C 51
SD.: Standard deviation; N/A: Not available; C39: Treacher Collin Syndrome: 40: Facial cleft & 0 = none; 1= intermittent or variable (occasionally heard on high pressure consonants and an error production needs to occur more than once for it to be documented of reliable error type) 2 = frequent or pervasive (heard on production of many high pressure consonants)
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Table 4. Prevalence of speech and language abnormalities (N=36)
Resonance Abnormal Language
Disorders
Voice disorders
delay
(mild+moderate (mild+moderate+severe) +severe)
18
% (95 % CI)
No.
50.00 (32.92, 67.08)
13
% (95 %CI)
No.
% (95 %CI)
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8.33 (1.75, 22.47)
No.
36.11 (20.82, 53.78)
11
30.56 (16.35, 48.11)
Number of articulation defects
Mean No
% (95 %CI)
% (95 %CI) (SD)
34
94.44 (81.34, 99.32)
12.69
10.62, 14.77
(6.15)
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Number of articulation defects: median = 12.5, 25th percentile=9.5, 75th percentile=17, 95% CI = 9.98, 15.02
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3
% (95 %CI)
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No.
SC
Understandability
ACCEPTED MANUSCRIPT Table 5. Prevalence of speech outcomes
Outcomes Percentage Understandability* 50.00 -Normal (0)
95% Confidence Interval 32.84, 67.16
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-Abnormal 38.89
22.16, 55.62
-Moderate (2)
2.78
0, 8.42
-Severe (3)
8.33
0, 17.82
63.89
47.41, 80.37
-Mild (1)
25.00
10.14, 39.86
-Moderate (2)
11.11
0.33, 21.90
-Severe (3) Voice*** -Normal (0)
69.44
51.89, 83.65
-Abnormal (1)
30.56
16.35, 48.11
Resonance** -Normal (0)
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-Abnormal
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-Mild (1)
*Rating of speech understandability – conversational Speech (21, 27) Normal: Intelligibility: the whole speech sample was always easy to understand; Mild: the whole
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speech sample was occasionally hard to understand; Moderate: the whole speech sample was often hard to understand; Severe: the whole speech sample was hard to understand most or all of the time
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**Rating of resonance disorders [21, 27] Normal: There was no perceptual evidence of cleft type speech and does not exceed nasality heard
in regional speech; Mild: Nasality exceeds regional speech nasality; Moderate: Hypernasality is perceived as pervasive and draws attention to itself and away from the message; Severe: Hypernasality is perceived as pervasive and interferes with speech understandability. ***Rating of voice disorders Normal: normal voice; Abnormality: any parameter of GIRBAS was scored as ≥ 1.
S1010-5182(16)30172-X
DOI:
10.1016/j.jcms.2016.08.004
Reference:
YJCMS 2456
To appear in:
Journal of Cranio-Maxillo-Facial Surgery
Received Date: 7 April 2016 Revised Date:
30 June 2016
Accepted Date: 3 August 2016
Please cite this article as: Prathanee B, Pumnum T, Seepuaham C, Jaiyong P, Five-Year Speech and Language Outcomes in Children with Cleft Lip-Palate, Journal of Cranio-Maxillofacial Surgery (2016), doi: 10.1016/j.jcms.2016.08.004. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Five-Year Speech and Language Outcomes in Children with Cleft Lip-Palate
Benjamas Prathanee PhD.*,
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Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand, e-mail address: [email protected] Tawitree Pumnum, BSc.
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Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
Cholada Seepuaham, BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
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Pechcharat Jaiyong, BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon
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Kaen, Thailand, 40002, e-mail address: [email protected]
*Corresponding author: Benjamas Prathanee, Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand, 40002, Tel. 66-43-363565, Fax: 66-43-202490, e-mail address: [email protected]
ACCEPTED MANUSCRIPT 1
Preschool Speech Outcomes in Children with Cleft Lip-Palate Benjamas Prathanee PhD.,
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Tawitree Pumnum BSc., Cholada Seepuaham BSc., Pechcharat Jaiyong BSc.
Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University,
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Khon Kaen, Thailand
1. Introduction
The worldwide incidence of cleft lip/palate was found to be between 0.11 and 2.65/1,000 live births (Hobbs et al., 2001, Chowchuen and Godfrey, 2003, Natsume and Tolarova 2006).
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Cleft lip and palate (CLP) is indeed a major public health concern in Thailand, where the incidence of cleft lip/palate is between 1.10 and 2.49/1,000 live births (Chuangsuwanich et al., 1998). CLP has several impacts on physical and psychological skills including nasolabial
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appearance, bone structure, swallowing, speech and language, hearing, dental, occlusion, delayed development, social growth, quality of life, as well as affecting individual and country
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economics. Interdisciplinary approaches from birth to 19 years of age are needed as a complete remedy.
Three important main outcomes of treatment for CLP are normal configuration, normal
speech functions and good quality of life. Surgery is needed for lip correction at age 3 months and palatoplasty at 1 year of age for relief of the stigmas of facial abnormality and to expect normal function of speech production. Residual speech and language problems after surgery,
ACCEPTED MANUSCRIPT 2
however, are of critical concern. The rates of speech and language abnormalities including delayed speech and language development were in the range of 16-19 %, articulation defects 2390% (Lohmander-Agerskov and Soderpalm, 1993, Schonweiler et al., 1999, Prathanee et al.,
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2013), resonance disorders 27-43 % (Schonweiler et al., 1999, Prathanee et al., 2013, Prathanee et al., 2014) and voice disorders 0.6-50 % ( Lohmander-Agerskov et al., 1995, Hamming et al., 2009, Robison and Otteson, 2011, Prathanee et al., 2013, Prathanee et al., 2014). Compensatory
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articulation disorders (CAD) are generally the most common problems for children with clefts.
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Children with CAD also presented with significant language delays (Pamplona et al., 2000). Children aged 4-8 years are in the transition phase from the pre-school to early school periods in which the development of cognitive skills and critical adaptation to others for living in society occurs (Child and Adolescent Health Service, 2013). Speech and language skills are crucial factors that affect the quality and appropriateness of character in school during this
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period. Based on Khon Kaen University Center’s protocol that is concerned with the balance of these outcomes, the schedules for labioplasty is at 3 months of age, palatoplasty and veloplasty at around 12 months of age are conducted to improve configuration, speech, language, hearing
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abilities and quality of life as early as possible. The investigation of speech and language
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outcomes at 5 years is necessary for further management and remedy planning to support adaptation in school, and feedback for reviewing the treatment protocols to improve clinical outcomes.
The aim of the present study was to investigate speech and language outcomes for
children with CLP aged around 5 years or who were in the transition phase from the pre-school to early school periods.
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2. Materials and methods After the research protocols were approved (August 7, 2009) by the Human Research
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Ethics Committee of Khon Kaen University in accordance with the Helsinki Declaration (HE521052), informed consents were obtained from the subjects’ legal representatives, and 38 children with CLP aged 4-7 years 8 months were recruited for this study. Children’s speech and
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language outcomes were assessed by the principal investigator and a qualified speech and
language pathologist (SLP) who was a research assistant and who had clinical experience related
then followed as shown below:
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to speech and language in cleft lip and palate children for more than 20 years. The protocol was
-
Oral examination and facial grimaces
-
Speech and language abilities with perceptual assessment of speech for clefts using
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the Thai Universal Parameters of Speech Outcomes for People with Cleft Palate and outcomes were recorded in case record forms that were developed for the standard test (Henningsson et al., 2008, Prathanee et al., 2011) included:
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Articulation: 2 rating scales. Each sound was evaluated by perceptual assessment. Score
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0=normal articulation; 1= abnormal articulation in any type of standard patterns for cleft palate speech (Henningsson, et al., 2008, Prathanee B et al., 2011). Resonance: 5 rating scales. Score 0=Normal: there was no perceptual evidence of cleft
type speech and does not exceed nasality heard in regional speech; 1 = Mild: Nasality exceeds regional speech nasality; 2 = moderate: hypernasality is perceived as pervasive and draws attention to itself and away from the message; 3 = severe: hypernasality perceived as pervasive and interferes with speech understandability.
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Voice: 2 scales. Voice assessment was performed by using the perceptual assessment or GIRBAS (Grade = overall impression, I: Instability or fluctuation of voice, R: Roughness, hoarseness, B: Breathiness, breathy voice, A: Asthenia or a weak voice or speaking with minimal
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air volume, S: Strain or forced or stressed voice). Each parameter was scored as 0= normal; 1=mild; 2=moderate; 3=severe. GIRBAS is a popular and reliable perceptual scale (Webb et al., 2004). Total score of GIRBAS: 0: normal voice; 1-18: abnormal voice. Score 0= normal; 1=
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abnormal voice).
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Intelligibility: 4 rating scale (0= normal: intelligibility or the whole speech sample was always easy to understand; 1= mild or the whole speech sample was occasionally hard to understand; 2= moderate or the whole speech sample was often hard to understand; 3= severe or the whole speech sample was hard to understand most or all of the time.
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Nasal emission/turbulence: 3 rating scales. Score 0=none; 1= intermittent or variable (occasionally heard on high pressure consonants and an error production needs to occur more than once for it to be documented of reliable error type); 2 = frequent or pervasive (heard on
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production of many high pressure consonants).
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- Language screening test (adapted Thai Early Language Milestone) (Lorwatanapongsa et al., 2011) and UTAH Test (Mecham and Jones, 1967)].
Statistical Analysis
The main outcomes were the numbers of children with CLP who had language delay, abnormal understandability, resonance abnormality, voice disturbance, and articulation defects.
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The oral examination described characteristics of oral configurations. Perceptual assessments were also scored as Resonance normal (0), abnormality if resonance was scored ≥1 hyponasality (-1), mild hypernasality (+1), moderate hypernasality (+2), severe hypernasality (+3) ; Voice as
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normal (0) and abnormal if any parameter of the voice was scored ≥ 1 (1: Understandability as normal (0), abnormality if understandability was scored ≥ 1 (1) (mild or occasionally hard to understand = 1, often hard to understand = 2, hard to understand most or all of the time = 3);
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Articulation defects as normal articulation development (0), at least 1 articulation error (1).
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Double entries and incorrect filing of data were corrected. Data analyses were performed by using numbers and percentages and analyzed by a Strata program.
3. Results
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Participants aged 4-8 years who were registered in the Khon Kaen University Center for Cleft Lip-Palate and Craniofacial Anomalies were enrolled in this study. Thirty-eight children with CLP were investigated for speech and language outcomes. General characteristics of
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children with CLP are displayed in Table 1.
Table 1
Children with CLP were 20 females and 18 males. Twenty-one children (55.26%) were
left CLP; 6 (15.79 %) were right CLP; and 11 (28.95 %) were bilateral CLP. With the exception of C 39 (Treacher Collins Syndrome) and C 40 (Facial cleft), seven of 36 children (19.44 %) presented with oronasal fistulas, two of them (5.26 %) had tongue-ties and 23 of 36 children
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(63.38 %) had malocclusions Class III. With the exception of the complicated or syndromic cases, C39 and C 40, data were
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described and analyzed based on total of 36 children. Five-year speech and language outcomes
nasometer and are displayed in Table 3.
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Table 2
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in children with CLP were assessed and are presented in Table 2. Resonance outcomes were by
Table 2 shows that 3 children had delayed speech and language development, 18 had an abnormality of understandability, 13 had resonance abnormalities or hypernasality, 11 had voice abnormality, and 34 had articulation defects. Thirty five of 36 children with CLP (97.22 %), with
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the exception of C28, incorrectly produced postalveolar trill or rolling R (/r/). The prevalence of articulation defects (34 of 36 children) with exception of /r/ was 94.44 %. C23 had only an error
Table 3
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of /r/.
There were 14 of 36 children with CLP (38.89 %) who had nasal emission or nasal
turbulence. Eight of 14 children (57.14 %) had hypernasality on perceptual assessment with nasal air emission or turbulence. Interestingly, 6 of 36 children (42.87 %) had normal resonance with some air emission or turbulence. Regarding speech and language disorders, prevalence rates are presented in Tables 4 and
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5. Table 4
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Articulation errors were the most common speech defects in children with CLP, and the average number of articulation errors among children with CLP was 12.69 % or a median 12.25 (Min= 0: Max =33), followed by the abnormality of understandability, hypernasality, and voice
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disorders.
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The prevalence of speech defects based on severity is shown in Table 5.
Table 5
Table 5 shows that the most common degree of abnormality was mild both in
4. Discussion
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these children.
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understandability and resonance disorders. Voice abnormality had the higher prevalence among
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Primary surgical protocol evaluation should be done at a patient age of approximately 5
years to reduce biases such as secondary surgeries or orthodontic treatment, and results are therefore obtained with minimal negative effects on facial growth (Dissaux, 2016). Optimal speech outcome is one of the most important functional goals of primary cleft surgery. Assessments of speech outcomes used in previous studies (Persson et al., 2002, Nyberg et al., 2010, Klinto et al., 2011, Britton, Albery et al., 2014, Dissaux, Grollemund et al., 2016)
ACCEPTED MANUSCRIPT 8
were used for estimation the main outcomes of this study. Data of 36 children with CLP, without heterogeneous cases (C39 and C40), are presented for assessment to plan for future appropriate treatment. The fistula rate was 19.44 % and was in the low- to mid-range compared to previous
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studies, range 2.4 % to 60% (Murthy et al., 2009, Stewart et al., 2009, Eberlinc and Kozelj, 2012, Hortis-Dzierzbicka et al., 2012). Besides the surgical technique, there might be other factors that affect the oronasal fistula rate including type and severity of clefts and experience of surgical
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skills (Murthy et al. 2009). These factors should be considered for surgical planning.
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For main outcomes, 3 children (7.89 %) had delayed speech and language development. This prevalence was low when compared to previous studies (range 13 -. 92 %) (Schonweiler et al., 1996, Vallino, Zuker et al., 2008, Ruiter et al., 2009, Rullo et al., 2009, Young et al., 2010, Prathanee et al., 2013). The early diagnosis and intervention program was effective for language stimulation and supports a previous finding (Ruiter, et al. 2009). Eighteen children with CLP (50
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%) had unclear speech or an understandability deviation. This was in the range of previous reports 44-63 % (Schuster et al., 2006, Normastura et al., 2008, Rullo et al., 2009). For resonance disorders, 14 of 38 children (36.11 %) had hypernasality and this was also in the range of
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previous reports of 20-43.30 % (Grunwell et al., 2000, Kummer, 2001, Sell et al., 2001,
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Prathanee, 2002, Kummer, 2008, Phua and de Chalain, 2008, Prathanee, 2012). This might validate that the intravelar veloplasty technique that is used in the KKU center was an effective surgical technique and seems to have less of a negative impact on maxillary growth and give good speech outcomes (Dissaux et al., 2016). In all children with CLP’s nasalance scores, Laying Hen (a load of every sound in Thai language) and My House (the load of both oral and nasal sounds), were out of normal range (more than mean ± 1SD) that agreed with perceptual assessment outcomes (mild and moderate hypernasality). This indicated that nasalance scores
ACCEPTED MANUSCRIPT 9
and perceptual assessments might be representative of each other. Overall, 11 of a total 36 children (30.56 %) had voice abnormalities and this was at a
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higher rate than previous studies, which ranged between 5.5-20.8 % (Hocevar-Boltezar et al., 2006, Hamming et al., 2009, Robison and Otteson, 2011, Prathanee B et al., 2013). Seven of 11 children (63.64%) with CLP had voice abnormalities with hypernasality (Table 2), however, the
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rest of them (4 of 11 or 36.36 %) had voice disorders and normal resonance by perceptual
assessment. This supported the theory that patients with CLP are at risk for voice disorders from
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both laryngeal hyperfunctions as a compensatory articulation mechanism for VPI ( Leder and Lerman, 1985, D'Antonio et al., 1988) and multifactorial variables related to voice abnormality. This occurs in clefts and includes gastroesophageal reflux diseases (Karkos et al., 2007, Fisichella, 2015) and might not relate to velopharyngeal insufficiency (Hamming et al., 2009).
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This needs to be confirmed by further study.
Cases of children with CLP who had a high rate of articulation errors with hypernasality of ≥ 12 articulation defects were found in 8 of 20 children (40.00 %), and the rest of them (12
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children or 60 %) had a high rate of articulation errors with normal resonance. This revealed that articulatory defects in CLP were not only caused by VPI, but might also be related to
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mislearning from incomplete kinesthetic patterns. For the other two patients, C39 and C40 (Treacher Collin Syndrome and Facial Cleft),
these patients had the largest number of articulation errors (21 and 18 sound errors) (Table 2), resulting in abnormal intelligibility. Even with palatoplasty for correction of velopharyngeal anatomy and good function that results in normal resonance (C39) and mild hypernasality (C40) without audible nasal emission or turbulence (Table 3), they might still have the limitation of
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oral structure from syndromic disease that is the cause of severe articulation errors and the need for long term speech therapy.
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There are limitations of speech services in Thailand, especially in the Northeast area composed of 21 cities and 21 million people, where the only tertiary level speech center and speech and language pathologists (SLP) are in the area where this study was done. There have
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been a few SLPs outside the KKU hospital center in the last 30 years. Therefore, most children with CLP received or no or little speech therapy, approximately one 30-minute session every 1-2
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months. The majority of them had not received continuous and long term speech therapy because 1) They could not afford expenses, e.g., living, transportation expenses, salary compensation etc., 2) It was a long distance to get speech services of 3-4 hours for a one way trip (Prathanee et al., 2006). Speech and language defects might be decreased if children with CLP could get appropriate speech therapy following the standard protocols. These data should provide feedback
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and stimulate more direct attention to the revision of protocols, including early speech and language intervention programs to solve compensatory articulation disorders on time and prevent long-term compensatory speech defects which need a longer and more difficult management.
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Successful speech camps or the Community-Based Speech Therapy Model are appropriate
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problem solving models (Pamplona et al., 1999, Pamplona et al., 2005, Prathanee et al., 2006, Prathanee et al., 2010, Prathanee et al., 2014). In addition, obligatory speech disorders caused from VPI that make incomplete kinesthetic patterns require early physical management and an early interventional speech program.
5. Conclusion
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Speech outcomes including articulation disorders, resonance disorders, voice abnormalities and understandability problems were of high prevalence. Early intervention programs and surgical techniques should be critically reviewed and the need further for
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prospective studies to improve speech outcomes for children with CLP should be pursued.
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Conflicts of interest:
Funding support:
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None
Cleft Lip/Palate and Craniofacial Abnormalities Center of Khon Kaen University
Acknowledgement
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Associate with “Tawanchai Foundation”.
The researchers thank the parents and children who participated in this project and Cleft
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Lip/Palate and Craniofacial Abnormalities Center of Khon Kaen University associated with “Tawanchai Foundation” for research and funding support. We would like to acknowledge Prof.
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James A. Will, for editing the MS via Publication Clinic KKU, Thailand.
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Table 1. Characteristics of children with clefts No.
Age
Gender
Diagnosis
Oral Examination
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(years & months)
- Lip contraction C 01
5; 10
Female
Lt.CLP
- Short uvula and active movement - Malocclusion Class III
Lt.CLP
C 04
4;5
Male
BCLP
C 05
5; 3
Male
Lt.CLP
C 06
5; 9
Female
Lt.CLP
- Bifid uvula and slight movement
Lt.CLP
- Short uvula and slight movement - Short uvula and active movement
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4
- Lip contraction
Female
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C 03
- Malocclusion Class III
- Bifid uvula and active movement - Malocclusion III
- Lip contraction
C 10
7; 8
Female
- Bifid uvula and active movement
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- Malocclusion III - Lip contraction
C 11
4; 3
Male
BCLP
- Oronasal fistula
- Bifid uvula and slight movement - Malocclusion III
C 20
C 21
6; 1
7; 1
Lt.CLP
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7; 1
Male
Male
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C 16
4; 4
AC C
C 12
Male
BCLP
- Lip contraction - Short uvula and active movement - Lip contraction and limited movement - Bifid uvula and slight movement - Malocclusion Class III - Lip contraction
Lt.CLP
- Bifid uvula and active movement - Malocclusion Class III - Lip contraction and limited movement
Female
BCLP
- Short uvula and active movement - Malocclusion Class II - Oronasal fistula
C 22
5; 10
Male
BCLP
C 23
5; 4
Female
Lt.CLP
- Lip contraction - Short uvula and slight movement - Lip contraction - Bifid uvula and slight movement
ACCEPTED MANUSCRIPT - Lip contraction C 24
6; 1
Female
Lt.CLP
- Bifid uvula and active movement - Oronasal fistula - Lip contraction
C 25
4; 6
Female
Lt.CLP and
- Short uvula and slight movement - Malocclusion Class II
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- Tongue tie
- Lip contraction C 26
5; 9
Male
Rt.CLP
- Short uvula and active movement - Malocclusion Class III
C 27
5; 9
Female
Lt.CLP
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- Lip contraction
- Oronasal fistula
- Bifid uvula and active movement
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- Alveolar Cleft
- Lip contraction
C 28
Male
5
Lt.CLP
- Short uvula and slight movement - Malocclusion Class III - Lip contraction
C 31
C 32
Lt.CLP
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Female
4
Female
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C 30
5; 3
5; 9
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C 29
4; 7
Female
BCLP
- Short uvula and slight movement - Oronasal fistula - Lip contraction - Short uvula and active movement - Malocclusion: open bite & missing upper teeth - Lip node/bumpy
BCLP
- Oronasal fistula - Short uvula and active movement - Malocclusion Class III - Lip contraction and limited movement
Male
BCLP
- Short uvula and active movement - Malocclusion Class III - Tongue Tie
C 33
5; 8
Female
Rt.CLP
C 34
4; 8
Male
Lt.CLP
- Lip contraction - Short uvula and active movement - Short uvula and active movement - Malocclusion Class III
ACCEPTED MANUSCRIPT - Lip contraction C 35
Female
4; 11
Lt.CLP
- Short uvula and active movement - Malocclusion Class III - Lip node or bumpy
C 36
Female
4; 10
Rt.CLP
- Bifid uvula and active movement - Malocclusion Class III
C 39
5
Male
Female
4; 10
Lt.CLP
BCLP and Treacher Collins Syndrome
- Bifid uvula and active movement - Malocclusion Class III
- Lip contraction and limited movement - Oronasal fistula - Bifid uvula and slight movement
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C 38
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- Right Alveolar Cleft
- Malocclusion Class III
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- Missing upper teeth - Lip contraction
C 40
4; 4
Male
Lt.CLP and Facial cleft
- Bifid uvula and slight movement - Malocclusion - Left Alveolar Cleft
C 42
4; 9
4; 2
C 44
4; 3
C49
AC C
C 48
Male
4; 4
Lt.CLP
Rt.CLP
Male
BCLP
Female
Rt.CLP
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C 43
C 47
Female
4; 11
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C 41
- Short uvula and active movement
- Bifid uvula and active movement - Malocclusion Class III - Right Alveolar Cleft - Lip contraction - Malocclusion Class III - Short uvula and active movement - Lip contraction
Male
BCLP
- Short uvula and active movement - Malocclusion Class III
4; 8
Male
Rt.CLP
5; 3
Male
Lt.CLP
- Lip contraction - Bifid uvula and active movement - Short uvula and active movement - Malocclusion Class III - Lip contraction
C 51
5; 4
Female
Lt.CLP
- Oronasal fistula - Bifid uvula and active movement
ACCEPTED MANUSCRIPT - Malocclusion Class III - Lip contraction C 52
4; 1
Female
Lt.CLP
- Bifid uvula and slight movement - Malocclusion Class III
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Lt CLP: left cleft lip and palate; Rt. CLP: Right cleft lip and palate; BCLP: bilateral cleft lip and palate.
ACCEPTED MANUSCRIPT Table 2. Five-year speech and language outcomes in children with CLP No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors
Pass
Normal
Normal
C 03
Pass
Normal
Normal
C 04
Pass
Mild
Normal
Abnormal
17- 2-0-0=19
C 05
Delayed
Mild
Mild
Normal
16-2-0-0=18
C 06
Pass
Normal
Normal
Normal
8-2-4-0= 14
C 10
Pass
Mild
Moderate
Abnormal
17-2-0-0=19
C 11
Delayed
Mild
Moderate
Normal
13-3-1-0=17
C 12
Pass
Normal
Normal
Normal
9-2-0-0=11
C 16
Pass
Mild
Mild
Abnormal
12-2-0-0= 14
C 20
Pass
Normal
Normal
Normal
7-2-2-0=11
C 21
Delayed
Normal
Mild
Normal
7-0-1-0=8
Pass
Normal
Normal
Normal
16-1-0-0=17
C 23**
Pass
Normal
Moderate
Normal
1-0-0-0=1
C 24
Pass
Normal
Mild
Normal
8-3-0-0=11
C 25
Pass
Normal
Normal
Normal
10-0-0-0=10
C 22
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Normal
14-2-0-1=17
Normal
15-3-4-1 =23
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C 01
AC C
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I-F-V-T*
ACCEPTED MANUSCRIPT No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors
Pass
Mild
Normal
C 27
Pass
Mild
Normal
C 28
Pass
Normal
Normal
C 29
Pass
Moderate
Moderate
Abnormal
17-0-0-0=17
C 30
Pass
Mild
Normal
Normal
11-3-0-0=14
C 31
Pass
Mild
Normal
Abnormal
8-2-1-0=11
C 32
Pass
Severe
Normal
Abnormal
16-1-0-0=17
C 33
Pass
Mild
Normal
Normal
10-1-0-0= 11
C 34
Pass
Mild
Normal
Normal
11-1-0-0=12
C 35
Pass
Normal
Mild
Abnormal
6-2-0-0=8
C 36
Pass
Normal
Mild
Normal
7-2-0-0=9
C 38
Pass
Normal
Normal
Normal
7-2-1-0=10
Pass
Mild
Normal
Normal
15-2-4-0=21
Pass
Severe
Mild
Abnormal
17-1-0-0=18
C 41
Pass
Normal
Normal
Normal
8-1-0-0=8
C 42
Pass
Normal
Normal
Normal
2-1-1-0=4
C 39
C 40
Normal
Normal
7-2-0-0=9
Normal
0-0-0-0=0
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12 -2-0-1=15
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C 26
AC C
I-F-V-T*
ACCEPTED MANUSCRIPT No.
Language
Understandability
Resonance
Voice
screening
No. articulation errors I-F-V-T*
Pass
Severe
Mild
Abnormal
C 44
Pass
Mild
Mild
C 47
Pass
Mild
Normal
C 48
Pass
Mild
Normal
Normal
11-2-1-0=14
C 49
Pass
Normal
Normal
Normal
11-2-1-0=14
C 51
Pass
Severe
Normal
Abnormal
11 -1-0 -0 =12
C 52
Pass
Normal
Mild
Abnormal
13-2-1-0 =16
18-5-7-3-0=33
Normal
17-2-2-0=21
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Abnormal
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C39: Treacher Collin Syndrome: 40: Facial cleft
* I: Initial sound; F: Final sound; V: Vowel; T: Tone
EP
**: Only articulation error was postalveolar trill or rolled R or rolling R (/r/)
AC C
11-1-1-0=13
RI PT
C 43
Table 3. Perceptual resonance assessment, nasalance scores and audible nasal emission/turbulence
ACCEPTED MANUSCRIPT
Audible emission/turbulence& Word Sentence
My house
Winter
Laying hen
Resonance
C 01
54
59
23
Normal
1
1
C 03
22
35
8
Normal
0
1
C 04
43
53
24
Normal
1
1
C 05$
52
60
38
Mild
1
1
C 06
37
52
20
Normal
0
0
C 10*
54
56
47
Moderate
0
0
C 11*
66
68
41
Moderate
1
1
C 12
31
50
16
Normal
0
0
C 16$
57
57
46
Mild
2
2
C 20
29
46
16
Normal
0
0
C 21
58
66
45
Mild
1
1
C 22
36
46
17
Normal
0
0
C 23
52
55
46
Moderate
1
1
C 24
60
70
58
Mild
0
0
C 25
48
52
22
Normal
0
0
39
54
21
Normal
0
0
SC
M AN U
TE D
EP
C 26
RI PT
Patient No.
41
58
17
Normal
0
0
C 28
39
56
31
Normal
0
0
C 29
40
41
35
Moderate
0
0
C 30
32
40
31
Normal
0
0
C 31
33
49
14
Normal
0
0
AC C
C 27
ACCEPTED MANUSCRIPT
C 32
52
74
Normal
Resonance
0
0
Audible emission/turbulence& Word Sentence
My house
Winter
Laying hen
31
50
9
C 34
38
46
72
Normal
0
0
C 35
46
51
31
Mild
2
2
C 36
55
62
31
Mild
0
1
C 38
28
49
9
Normal
0
0
C 39
40
37
22
Normal
0
0
C 40
44
42
C 41
38
57
C 42
28
46
C 43
44
62
C 44
37
C 47
48
C 48
42
C 49
0
M AN U
SC
RI PT
Normal
0
36
Mild
0
0
18
Normal
0
0
14
Normal
1
1
51
Mild
0
0
53
44
Mild
0
0
54
36
Normal
0
0
54
30
Normal
1
2
Normal
0
0
TE D
C 33
EP
Patient No.
64
N/A
N/A
N/A
36
46
24
Normal
0
1
C 52
40
47
23
Mild
0
1
Mean (SD.)
42.76 (10.76)
52.19 (8.06)
30.81 (16.36)
95 % Confident Interval
39.17- 46.34
49.50- 54.88
25.35-36.27
Norm{Prathanee, 2003 #123}
35.6 (5.9)
51.1 (6.4)
14.3 (5.8)
AC C
C 51
SD.: Standard deviation; N/A: Not available; C39: Treacher Collin Syndrome: 40: Facial cleft & 0 = none; 1= intermittent or variable (occasionally heard on high pressure consonants and an error production needs to occur more than once for it to be documented of reliable error type) 2 = frequent or pervasive (heard on production of many high pressure consonants)
ACCEPTED MANUSCRIPT
RI PT
Table 4. Prevalence of speech and language abnormalities (N=36)
Resonance Abnormal Language
Disorders
Voice disorders
delay
(mild+moderate (mild+moderate+severe) +severe)
18
% (95 % CI)
No.
50.00 (32.92, 67.08)
13
% (95 %CI)
No.
% (95 %CI)
M AN U
8.33 (1.75, 22.47)
No.
36.11 (20.82, 53.78)
11
30.56 (16.35, 48.11)
Number of articulation defects
Mean No
% (95 %CI)
% (95 %CI) (SD)
34
94.44 (81.34, 99.32)
12.69
10.62, 14.77
(6.15)
TE D
Number of articulation defects: median = 12.5, 25th percentile=9.5, 75th percentile=17, 95% CI = 9.98, 15.02
EP
3
% (95 %CI)
AC C
No.
SC
Understandability
ACCEPTED MANUSCRIPT Table 5. Prevalence of speech outcomes
Outcomes Percentage Understandability* 50.00 -Normal (0)
95% Confidence Interval 32.84, 67.16
RI PT
-Abnormal 38.89
22.16, 55.62
-Moderate (2)
2.78
0, 8.42
-Severe (3)
8.33
0, 17.82
63.89
47.41, 80.37
-Mild (1)
25.00
10.14, 39.86
-Moderate (2)
11.11
0.33, 21.90
-Severe (3) Voice*** -Normal (0)
69.44
51.89, 83.65
-Abnormal (1)
30.56
16.35, 48.11
Resonance** -Normal (0)
TE D
M AN U
-Abnormal
SC
-Mild (1)
*Rating of speech understandability – conversational Speech (21, 27) Normal: Intelligibility: the whole speech sample was always easy to understand; Mild: the whole
EP
speech sample was occasionally hard to understand; Moderate: the whole speech sample was often hard to understand; Severe: the whole speech sample was hard to understand most or all of the time
AC C
**Rating of resonance disorders [21, 27] Normal: There was no perceptual evidence of cleft type speech and does not exceed nasality heard
in regional speech; Mild: Nasality exceeds regional speech nasality; Moderate: Hypernasality is perceived as pervasive and draws attention to itself and away from the message; Severe: Hypernasality is perceived as pervasive and interferes with speech understandability. ***Rating of voice disorders Normal: normal voice; Abnormality: any parameter of GIRBAS was scored as ≥ 1.