|Year : 2021 | Volume
| Issue : 2 | Page : 103-110
Cleft lip and palate: Relationship between phonological awareness and audiological characteristics in children
Mônica Elisabeth Simons Guerra1, Vanessa Magosso Franchi2, Beatriz Cavalcanti Albuquerque Caiuby Novaes2, Mariana Lopes Favero2, Sulene Pirana3
1 Departament of Speech Therapy, Pontifical Catholic University of São Paulo, São Paulo, Brazil
2 Audiology, Pontifical Catholic University of São Paulo, São Paulo, Brazil
3 Department of otorhinolaryngology, University of São Paulo, São Paulo, Brazil
|Date of Submission||02-Dec-2020|
|Date of Acceptance||18-Dec-2020|
|Date of Web Publication||7-Jun-2021|
Dr. Mônica Elisabeth Simons Guerra
Department of Speech Terapy, Pontifical Catholic University of São Paulo, São Paulo
Source of Support: None, Conflict of Interest: None
Context: Cleft lip and palate (CLP) stands out due to its esthetic-functional impact and its high incidence. Phonological awareness involves skills related to the structure of language, and children with CLP may experience impairment in these skills due to auditory impairment resulting from otitis media and fluctuating hearing loss. Aim: The aim of this study was to correlate phonological awareness with audiological characteristics in children with CLP. Materials and Methods: Thirty children, 13 (43%) with cleft palate (CP) and 17 (57%) with CLP from 6 to 9 years old, were evaluated at a specialized center in Brazil to determine their phonological awareness skills and were classified into low, medium, and high skills categories. Phonological awareness was related to the speech reception threshold (SRT) at two time points: at 3 years old and at the time of data collection. Results: The study population was homogeneous in terms of age, sex, and socioeconomic status. In terms of phonological awareness skills, 8 children (26.7%) were classified as having low skills, 9 (30%) were classified as medium, and 13 (43.3%) were classified as high. Low phonological awareness skills were associated with worse mean SRT at 3 years old (mean = 34.4 dB, standard deviation [SD] = 11.2, P = 0.046) and at the time of data collection (mean = 22.2 dB, SD = 17.2). Conclusion: The association of phonological awareness with SRT suggests that prolonged or transient hearing loss can lead to changes in phonological skills and auditory processing.
Keywords: Cleft lip and palate, language, otitis media effusion, phonological awareness
|How to cite this article:|
Guerra ME, Franchi VM, Novaes BC, Favero ML, Pirana S. Cleft lip and palate: Relationship between phonological awareness and audiological characteristics in children. J Cleft Lip Palate Craniofac Anomal 2021;8:103-10
|How to cite this URL:|
Guerra ME, Franchi VM, Novaes BC, Favero ML, Pirana S. Cleft lip and palate: Relationship between phonological awareness and audiological characteristics in children. J Cleft Lip Palate Craniofac Anomal [serial online] 2021 [cited 2021 Sep 28];8:103-10. Available from: https://www.jclpca.org/text.asp?2021/8/2/103/317789
| Introduction|| |
Children with cleft lip and palate (CLP) may have great variability in the language that can be the result of a complex interaction between biological and environmental factors.,,, Before 1 year old, otitis media effusion (OME) will occur at least once in 90% of children with cleft palate (CL) and has both short- and long-term negative impacts on language development and quality of life;,,,,,,, therefore, some doctors routinely perform prophylactic insertion of ventilation tube at the time of palate repair.,,,,,,,,
Many studies have analyzed the impact of the effect of clefts on language development.,,,,,,,, Phonological awareness is a skill involving recognition and manipulation of linguistic sounds beyond their meanings through reflection on representation of language components, and it has clear importance in acquisition of reading and writing abilities.,,,,,, This cross-sectional observational study posed a question as to whether history of recurrent otitis media with auditory threshold shifts in the 1st year of age of children with CLP interferes with phonological awareness skills later in life.
| Materials and Methods|| |
This observational cross-sectional study was approved by the institution's Research Ethics Committee for a number 2,403,672.
Participants and place
The children were selected according to medical record data from a specialized health center in the state of São Paulo (Brazil) since birth.
The sample consisted of 30 children with CLP; in order to keep the study more homogeneous, only children with malformation of the palate were selected. According to medical records, following the modified Spina classification, the children were divided into two subgroups: 13 (43%) children with CP and 17 (57%) children with CLP (including 6 children with bilateral CLP, 5 children with right unilateral CLP, and 6 children with left unilateral CLP). All children (100%) had a palate corrected by the surgical procedure.
The children were evaluated in a quiet room with a parent or legal guardian, by a phoniatric/ENT doctor. Information about age, socioeconomic level, sex, history of OME, physical examination (otoscopy), children's behavior, and school performance was obtained on semi-structured interview with parents, physical examination (ENT) and medical records. The children were aged 6–9 years (mean = 8 years and 2 months); 14 children (47%) were male and 16 (53%) were female. All were enrolled in a regular school in elementary school, since preschool (between 3 and 4 years old). Elementary education is one of the levels of basic education in Brazil. Elementary education is compulsory, free (in public schools), and serves children from 6 years of age. The school year runs from February over a calendar year.
The socioeconomic analysis is based on the methodology of the socioeconomic classification proposed by the author Graciano. In relation to the socioeconomic level, most families belonged to low class (80%).
According to semi-structured interview with parents, described in the Appendix 1, 13 children (43.3%) had inadequate school performance, 13 (43.3%) had difficulties in the school environment, and 15 (50%) had behavior complaints. In this study, a semi-structured interview was chosen with the parents instead of written questionnaire. In low-income population, parents may have limited education and written questionnaires are rarely used.
All children were followed up by an audiologist, a speech therapist, and a psychologist for periodic evaluation. Therapy was recommended according to their needs and was undertaken at the same service, where the present study took place. Twenty-five (83.3%) children in the study underwent speech therapy in a period ranging from 6 months to 84 months.
The children and their guardians received written and verbal clarification regarding the study objectives and possible risks and agreed to participate in the study, signing the terms of consent and assent. The exclusion criteria were sensorineural hearing loss, intellectual disability, moderate-to-severe visual impairment, or associated syndromes, according to medical record data.
Test of phonological awareness
The Test of Phonological Awareness by Oral Production, developed in Brazil in the Portuguese language for children aged 3–14 years old, was applied.
Each child receives instructions for each subtest, consisting of two examples and four exercises. The Test of Phonological Awareness consists of the following skills: (1) syllabic and phonemic synthesis. The child must hear the word syllables and phonemes, and then identify the word; (2) rhyme recognition. The child identifies, in a series of three words, two words that end with the same sound; (3) alliteration awareness. He/she identifies, in a series of three words, two words that start with the same sound; (4) syllabic and phonemic segmentation. He/she must hear a word and then separate it into syllables and phonemes; (5) syllabic and phonemic manipulation. The child must subtract or add syllables or phonemes of words saying which word is formed; (6) syllabic and phonemic transposition. He/she must invert the syllables and phonemes of words saying which word is formed.
One score was noted for each correct exercise, with a maximum score of 40. The total scores were listed in a table to identify the standard score, according to age and Brazilian school graduation. Standard scores below 84 were classified in low category, between 85 and 114 in medium category, and above 115 in high category.
The examination chosen in the study for hearing assessment in children was speech reception threshold (SRT), determined in two moments, at time of collection and at 3 years of age through analysis of medical record. Speech recognition tests are responsible for confirming tonal thresholds and can be more interesting for the child as they keep him alert for sounds of greater intensity. From the age of 8 months, when the child begins to recognize names and simple commands, it is possible to perform the search for SRT using simple repeated phrases from the child's repertoire. The SRT corresponds to the lower intensity that the individual can identify; 50% of the familiar words, disyllables and trisyllables, which are presented to him and usually these values are compatible with the average of the hearing thresholds 500 Hz, 1000 Hz, and 2000 Hz. It was considered SRT altered above 15 dB in the assessment of children.
In the analysis of the auditory skills tests, the association of phonological awareness and the following variables were initially verified: age (analysis of variance [ANOVA]), sex (likelihood ratio test), socioeconomic level (likelihood ratio test), school performance (reason test likelihood), SRT at 3 years old and at the time of data collection (Kruskal–Wallis test), and type of fissure (likelihood ratio test).
Variables with a P value lower than 0.200 in this analysis were used as explanatory variables in a multinomial logistic regression model, with phonological awareness as a response variable. These models allowed us to evaluate the combined effect of explanatory variables on phonological awareness. These models allow assessing the joint effect of the explanatory variables on phonological awareness. In the hypothesis tests, a significance level of 0.05 was set.
| Results|| |
Characterization of the sample
First, there was no difference in the mean age (P = 0.618) and the means of the probability distribution of sex (P = 0.431) and socioeconomic level (P = 0.580) between the two cleft subgroups: children with CP and children with CLP. Seventeen (56.7%) children had adequate school performance according to parents' report, and 13 (43.3%) children had inadequate school performance. There was no significant difference in the percentages of children with adequate school performance in two cleft subgroups (P = 0.785).
In according to medical record data, all children had a history of OME in the 1st year of age. Nineteen children (63.3%) underwent tympanotomy for the placement of ventilation tubes before the 3rd year of age. There was no significant difference in the distribution of the number of tympanostomies between the two cleft subgroups (P = 0.379). At the time of collection, 21 (70%) children showed changes in otoscopy characterized by tympanic membranes with retraction, perforation, tympanosclerosis, and insertion of the ventilation tube.
Performance on the phonological awareness test
On the phonological awareness test, according to age and school graduation, 8 (26.7%) children had scores that placed them in the low category, 9 (30%) children placed in the medium category, and 13 (43.3%) children placed in the high category.
There was no difference in the distributions of probability of phonological awareness based on sex (P = 0.239), socioeconomic level (P = 0.812), or cleft type (P = 0.924) (likelihood ratio test). Age means were not the same among the phonological awareness categories (P = 0.001) (ANOVA) [Table 1]. The results shown in [Table 1] were obtained following the analysis used by Tukey's method. The age mean of the high category was higher than that of the other categories, and there was no significant difference in the means between the low and medium categories.
|Table 1: Descriptive summary of age (months) by phonological awareness category in children with cleft lip and palate|
Click here to view
Most of the children with inadequate school performance had phonological awareness in the low category (53.8%), while most children with satisfactory school performance had phonological awareness in the high category (58.8%). The distributions of probability of phonological awareness ability in the two categories of school performance were not the same (P = 0.009) (likelihood ratio test) [Table 2]. Furthermore, a qualitative inspection of the study data related to the phonological skills that were part of the phonological awareness test showed that transposition, manipulation, phonemic synthesis, and phonemic segmentation were the most impaired skills.
|Table 2: Frequency distributions and percentages of phonological awareness by school performance in children with cleft lip and palate|
Click here to view
Individual and average values of the SRTs in each category of phonological awareness are shown in [Table 3] and [Table 4]. The mean SRTs at 3 years were worse in the low phonological awareness category than in the medium and high categories. The distributions of the SRT were not all the same among the three phonological awareness categories (P = 0.046), i.e., there was a significant association between SRT at 3 years old and later phonological awareness (Kruskal–Wallis test). On the other hand, at the time of data collection of phonological awareness skills, the mean SRTs were also worse in the low phonological awareness category (mean: 22.2 dB, standard deviation = 17.2). However, no significant difference was detected in the distributions of SRT among the three phonological awareness categories (P = 0.276) (Kruskal–Wallis test). Worse SRT values in the 1st year of age seem to impact phonological awareness skills at school age.
|Table 3: Descriptive summary of the mean speech reception threshold (dB) in both ears at 3 years old in children with cleft lip and palate|
Click here to view
|Table 4: Descriptive summary of the mean speech reception threshold (dB) in both ears at the time of data collection of age in children with cleft lip and palate|
Click here to view
The variables that in assessment of association with phonological awareness presented above with P < 0.200, were considered as explanatory variables in the adjustment of a multinomial logistic regression model, in which phonological awareness is response variable. This model allows to evaluate the joint effect of these variables on phonological awareness.
Thus, explanatory variables were considered: age, school performance, and SRT at 3 years old. The low category of phonological awareness was considered as the reference category for the response variable. Among the explanations, school performance is a qualitative variable and the category “;no” (inadequate school performance) was chosen as a reference [Table 5].
|Table 5: Nominal logistic regression: Phonological awareness versus age, school performance, and speech reception threshold at 3 years old|
Click here to view
As phonological awareness had three response categories (low, medium, and high), the effects of the explanatory variables were first presented in the logarithm of the odds of phonological awareness being in the high category compared with the low category. In the second analysis, the effect of the explanatory variables was presented in the logarithm of the odds of phonological awareness being in the medium category compared with the low category. The interpretation of these results is as follows:
Chance of phonological awareness being in the high category compared to the low category. The P value associated with age was 0.031, and the coefficient of this variable was positive. Thus, there is evidence that the odds of being in the high category relative to the low category increase with age when the remaining variables were kept constant in the model. The P values associated with school performance and SRT at 3 years were, respectively, 0.069 and 0.09, which are >0.05 but <0.10; i.e., it can be said that these variables had a marginal effect on the odds of being in the high category. The lack of statistical power can be attributed to the moderate sample size. The negative coefficient of SRT at 3 years indicates that the likelihood of being in the high category decreases with increasing SRT at this age.
| Discussion|| |
CLP has important characteristics: the complexity of language problems and the high incidence in the Brazilian and global population.,,, Thus, a comprehensive clinical approach, rather than a focus on physical anomaly, will influence several aspects in the child's development., The present study evaluated children with CLP and CP seen at a reference center in the state of São Paulo (Brazil) that specializes in multidisciplinary functional and esthetic rehabilitation from birth to adulthood. These children were submitted to medical monitoring and therapy to deal with their difficulties; therefore, the group of children studied represents children who received the recommended care in the expected time. Much of the results observed regarding children's performance, refer to language skills trained in speech therapy.
Our goal with the study was to discuss whether history of OME with auditory threshold shifts in the 1st year of age of CP children interferes with phonological awareness skills later in life.
An association was found between the phonological awareness categories and the SRT in an analysis of the mean SRT at two time points: 3 years old and the time of data collection. This relationship was significant for the SRT at 3 years old: the children with the worst mean SRTs at 3 years old had a high likelihood of phonological awareness skills in the low category, while those with the best mean SRTs had a greater likelihood of having phonological awareness skills in the high category. In the present study, the results of phonological awareness skills tests showed a significant association with school performance.
The SRT data reflect the audiological history of children with CLP, suggesting that transient or prolonged hearing loss may interfere with the child's opportunities to process speech stimuli and consequently may lead to varying degrees of phonological deficit, language problems,,,,,,,, and school problems,,,,,,, even in cases of children that were accompanied by speech therapy.
The sample comprised two subgroups of children with palate malformation and consequent function failure in the muscles that open the Eustachian tube More Details.,,,,,,,,, The subgroups, which were classified by type of cleft, were homogeneous in terms of age, sex, and socioeconomic level.
Most of the children with inadequate school performance had impairment in phonological awareness skills, while most children with satisfactory school performance had better results in the performance of the phonological awareness test (high category). In the process of learning to read and write, children exercise the skill of thinking about words (phonological awareness), and they become able to write by constructing their internal speech and systematically increasing the structure of their ability to reason in words.,,,,,,
Carroll and Breadmore (2018),, analyzing a group of poor readers, compared controls matched for reading level and children with a history of otitis media. Children with history of otitis media had phonological awareness score below the level of reading age matched controls but showed only small delays in their literacy and morphological awareness. Further analysis suggested that this weakness was primarily in segmenting and blending phonemes, implying that goals of intervention should be carefully chosen. The present study analyzed children with a history of otitis media and different degrees of transient hearing. In this sense, the results seem to suggest that impaired hearing in the 1st year of age can have consequences for the development of linguistic skills.
Older children in the study 9 years approximately, had the best results in phonological awareness. Two factors contributed. First, most children underwent speech therapy at referral service developing speech and language skills. Another important thing is phonological training in the 1st year of elementary school for reading and writing. The decoding process prepares the child to reflect on sounds to identify similar sounds and thus gradually building mental lexicon that results in successive nonlinear neurological maturation.,,
This study had some limitation in terms of population and sample size.
The population was consisted of children with CLP, who attended at a specialized health center by an interdisciplinary team since birth. Thus, children had different clinical conditions directly related to language functional characteristic heterogeneity. This influenced the authors' decision in not having a control group, therefore, using data from the literature for discussion. As the objective of this study was related to audiological characteristics with the results of analysis of phonological awareness test, a group with cleft lip was not used in comparison with other types of cleft.
The inclusion and exclusion criteria, identified through clinical history and analysis of data from medical records, may have contributed to the limitation of the sample size and the formation of small subgroups of fissures. What may have interfered in the statistical interpretation of the analysis are some variables with phonological awareness.
However, according to the authors, this study may contribute to health professionals who do not have many patients CLP, such as in a specialized center. According to the literature, the occurrence of middle ear changes in the 1st year of age may have consequences in future skills involved in auditory processing and language development. The study contributes to a greater understanding on the subject and to assist future research on language disorders with patients with CLP.
| Conclusion|| |
In this study, the level of phonological awareness skills in children with CLP and CP had a significant relationship with the SRT at 3 years old, which suggests that hearing loss in the 1st year of age, due to otitis media, causing changes in the auditory threshold, can have consequences on the development of phonological skills in the school period.
Financial support and sponsorship
M.E.S.G received a scholarship from the Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES) (Finance code: 88887.151893/2017-00).
Conflicts of interest
There are no conflicts of interest.
| Appendix 1|| |
| References|| |
Freitas JA, Neves LT, Almeida AL, Garib DG, Trindade-Suedam IK, Yaedú RY, et al
. Rehabilitative treatment of cleft lip and palate: Experience of the hospital for rehabilitation of craniofacial anomalies/USP (HRAC/USP)-Part 1: Overall aspects. J Appl Oral Sci Bauru 2012;20:9-15.
Pamplona MD, Ysunza PA. Total immersion speech camps for patients with cleft palate. J Cleft Lip Palate Craniofac Anomal 2017;4:132-8. [Full text]
Tengroth B, Hederstierna C, Neovius E, Flynn T. Hearing thresholds and ventilation tube treatment in children with unilateral cleft lip and palate. Int J Pediatr Otorhinolaryngol 2017;97:102-8.
Menegueti KI, Mangilli LD, Alonso N, Andrade CR. Perfil da fala de pacientes submetidos à palatoplastia primária. CoDAS 2017;29:e20160146.
Amaral MI, Martins JE, Santos MF. Estudo da audição em crianças com fissura labiopalatina não-sindrômica. Braz J otorhinolaryngol 2010;76:164-71.
Kuo CL, Tsao YH, Cheng HM, Lien CF, Hsu CH, Huang CY, et al
. Grommets for otitis media with effusion in children with clef palate: A systematic review. Pediatrics 2014;134:983-94.
Rieu-Chevreau C, Lavagen N, Gbaguidi C, Dakpé S, Klopp-Dutote N, Page C. Risk of occurrence and recurrence of otitis media with effusion in children suffering from cleft palate. Int J Pediatr Otorhinolaryngol 2019;120:1-5.
Klopp-Dutote N, Kolski C, Strunski V, Page C. Tympanostomy tubes for serous otitis media and risk of recurrences. Int J Pediatr Otorhinolaryngol 2018;106:105-9.
Flynn T, Möller C, Jönsson R, Lohmander A. The high prevalence of otitis media with effusion in children with cleft lip and palate as compared to children without clefts. Int J Pediatr Otorhinolaryngol 2009;73:1441-6.
Phua YS, Salkeld LJ, de Chalain TM. Middle ear disease in children with cleft palate: Protocols for management. Int J Pediatr Otorhinolaryngol 2009;73:307-13.
Rosenfeld RM, Shin JJ, Schwartz SR, Coggins R, Gagnon L, Hackell JM. et al
. Clinical practice guideline: Otitis media with effusion (update). Otolaryngol Head Neck Sugery 2016;154:SI-41.
Schönweiler B, Schönweiler R, Schmelzeisen R. Language development in children with cleft palate. Folia Phoniatr Logop 1996;48:92-7.
Tunçbilek G, Ozgür F, Belgin E. Audiologic and tympanometric findings in children with cleft lip and palate. Cleft Palate Craniofac J 2003;40:304-9.
Valtonen H, Dietz A, Qvarnberg Y. Long-term clinical, audiologic, and radiologic outcomes in palate cleft children treated with early tympanostomy for otitis media with effusion: A controlled prospective study. Laryngoscope 2005;115:1512-6.
Robinson PJ, Lodge S, Jones BM, Walker CC, Grant HR. The effect of palate repair on otitis media with effusion. Plast Reconstr Surg 1992;89:640-5.
Maheshwar AA, Milling MA, Kumar M, Clayton MI, Thomas A. Use of hearing aids in the management of children with cleft palate. Int J Pediatr Otorhinolaryngol 2002;66:55-62.
Shaw R, Richardson D, McMahon S. Conservative management of otitis media in cleft palate. J Craniomaxillofac Surg 2003;31:316-20.
Zumach A, Gerrits E, Chenault MN, Anteunis LJ. Otitis media and speech-in-noise recognition in school-aged children. Audiol Neurootol 2009;14:121-9.
Chapman KL. The relationship between early reading skills and speech and language performance in young children with cleft lip and palate. Cleft Palate Craniofac J 2011;48:301-11.
Hardin-Jones M, Chapman KL. Early lexical characteristics of toddlers with cleft lip and palate. Cleft Palate Craniofac J 2014;51:622-31.
Devolver IJ. Language and reading dysfunction in boys with isolated cleft lip and/or palate: A relationship to abnormal structural and functional connectivity in the brain (tese). Iowa: University of Iowa Libraries; 2015.
Young SE, Purcell AA, Ballard KJ. Expressive language skills in Chinese Singaporean preschoolers with nonsyndromic cleft lip and/or palate. Int J Pediatr Otorhinolaryngol 2010;74:456-64.
Pamplona MD, Ysunza PA. Language proficiency in children with cleft palate. Int Arch Commun Disord 2018;1:1-7.
Fitzsimons KJ, Copley LP, Setakis E, Charman SC, Deacon SA, Dearden L, et al
. Early academic achievement in children with isolated clefts: A population-based study in England. Arch Dis Child 2018;103:356-62.
Lancaster HS, Lien KM, Chow JC, Frey JR, Scherer NJ, Kaiser AP. Early speech and language development in children with nonsyndromic cleft lip and/or palate: A meta-analysis. J Speech Lang Hear Res 2020;63:14-31.
Carroll JM, Breadmore HL. Not all phonological awareness deficits are created equal: Evidence from a comparison between children with otitis media and poor readers. Dev Sci 2018;21:e12588.
Carroll JM, Snowling MJ, Hulme C, Stevenson J. The development of phonological awareness in preschool children. Dev Psychol 2003;39:13-23.
Seabra AG, Trevisan BT, Capovilla FC. Teste infantil de nomeação. In: Seabra AG, Dias NM, eds. Avaliação neuropsicológica cognitiva: linguagem oral. Vol 2. São Paulo: Memnon; 2012. p. 54-86.
Liberman LY, Shankweiler D, Liberman AM. The alphabetic principle and learning to read. In: Liberman IY, Shankweiler D. Phonology and reading disability: Solving the reading puzzle. Michigan: Ann Arbor the University of Michigan Press; 1989. p. 1-34.
Anthony JL, Francis DJ. Development of phonological awareness. Curr Directions Psychol Sci 2005;14:255-9.
Seabra AG, Capovilla FC. Teste de consciência fonológica por produção oral. In: Seabra AG, Dias NM, eds. Avaliação neuropsicológica cognitiva: Linguagem oral. Vol 2. São Paulo: Memnon; 2012. p. 117-22.
Capellini SA, Cunha VOC. Desempenho de escolares de 1 a 4 série do ensino fundamental nas provas de habilidades metafonológicas e de leitura – PROHMELE. Rev Soc Bras Fonoaudiol 2009;14:56-68.
Trindade JE, Silva Filho OG. Fissuras labiopalatinas-uma abordagem interdisciplinar. São Paulo: Ed. Santos; 2007.
Graciano MI. Estudo Socioeconômico: Um instrumento técnico-operativo. São Paulo: Veras; 2013.
Momensohen-Santos TM, Russo IC. The practice of clinical audiology. Rio de Janeiro 2015 p. 291-310.
Hosmer DW, Lemeshow S. Applied logistic regression. New York: John Wiley and Sons Inc; 2000.
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]