The Cleft Palate–Craniofacial Journal 52(1) pp. 70–81 January 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

ORIGINAL ARTICLE Spelling Processes of Children With Nonsyndromic Cleft Lip and/or Palate: A Preliminary Study Karen Shi Mei Lee, M.Sc., B.A., Selena Ee-Li Young, Ph.D., M.Sc., B.App.Sc., Susan Jane Rickard Liow, Ph.D., Dip.Clin.Psych., B.Sc., Alison Anne Purcell, Ph.D., M.App.Sc., B.App.Sc. Objective: To compare the cognitive-linguistic processes underlying spelling performance of children with cleft lip and/or palate with those of typically developing children. Design: An assessment battery including tests of hearing, articulation, verbal short-term and working memory, and phonological awareness, as well as word and nonword spelling, was administered to both groups. Participants: A total of 15 children with nonsyndromic cleft lip and/or palate were casematched by age and sex to 15 typically developing children. The children were aged between 6 and 8 years and were bilingual, with English the dominant language. Results: Wilcoxon signed-rank tests revealed that the performance of children with cleft lip and/or palate was significantly poorer on phoneme deletion and nonword spelling (P , .05) compared with typically developing children. Spearman correlation analyses revealed different relationships between the cognitive-linguistic and spelling measures for the cleft lip and/or palate and typically developing groups. Conclusions: Children with cleft lip and/or palate underachieve in phonological awareness and spelling skills. To facilitate early intervention for literacy problems, speech-language pathologists should routinely assess the cognitive-linguistic processing of children with cleft lip and/or palate, especially phonological awareness, as part of their case management protocols. KEY WORDS:

cleft, nonword, phonological awareness, spelling, verbal memory

development of children with CL/P has focused on reading disabilities (e.g., Broder et al., 1998; Richman and Eliason, 2001; Richman and Ryan, 2003; Richman et al., 2005; Collett et al., 2010a; Collett et al., 2010b). Although spelling skills are critical for fluent writing and academic success (McCutchen et al., 2008), there is relatively little empirical work on the spelling performance of the CL/P population and their underlying cognitive-linguistic processing. Good spelling ability, like good reading ability, usually involves sublexical (phonological) processing, particularly phoneme awareness (Treiman, 2000; Gillon, 2004), and depends on speech (Treiman et al., 1997). Of note, in a study of 5-year-olds, Jalil and Rickard Liow (2008) provided strong evidence that beginners’ spellings are based on phonological redintegration of their own speech-based representations, not those of parents or teachers. Children with CL/P are known to be at high risk for speech and resonance problems, such as compensatory speech patterns, hypernasality, and nasal emission (Harding and Grunwell, 1996; Kuehn and Moller, 2000). In addition, these children often have associated abnormalities of the dental arch, missing or misplaced teeth, and fistulas (openings between the oral and nasal cavities; Kuehn and Moller, 2000). The likelihood of distortions, or any delay in speech development would therefore put children with CL/ P at risk for difficulty with early spelling and writing skills.

Children with cleft lip and/or palate (CL/P) are reported to underachieve in many school settings (Richman and Eliason, 1986; Buckenberger, 1990; Broder et al., 1998) as a result of their reported speech, language, and memory deficits (Grunwell and Russell, 1988; Jocelyn et al., 1996; Kuehn and Moller, 2000; Peterson-Falzone et al., 2001; Morris and Ozanne, 2003). Research profiling the literacy

Ms. Lee is Speech Therapist, Department of Plastic, Reconstructive, and Aesthetic Surgery, KK Hospital, Singapore. Dr. Young is Lecturer, Division of Graduate Medical Studies, Yong Loo Lin School of Medicine, National University of Singapore, and Head and Senior Principal Speech Therapist, Department of Plastic, Reconstructive, and Aesthetic Surgery, KK Hospital, Singapore, and Department of Otolaryngology, National University Health System, Singapore. Dr. Rickard Liow, Associate Professor, Department of Otolaryngology, is Programme Director of the MSc (Speech and Language Pathology) at the National University of Singapore. Dr. Purcell is Course Director, Discipline of Speech Pathology, University of Sydney, Sydney, Australia, and Certified Practicing Speech Pathologist, Sydney, Australia. Paper presented at Speech Pathology Australia 2012 National Conference, Communicate: Our Natural State, June 24–27, 2012, in Hobart, Tasmania, Australia. Submitted May 2013; Revised September 2013; Accepted December 2013. Address correspondence to: Karen Lee Shi Mei, Department of Plastic, Reconstructive, and Aesthetic Surgery, Kandang Kerbau Women’s and Children’s Hospital, 100 Bukit Timah Road, Singapore 229899. E-mail [email protected]. DOI: 10.1597/13-120 70

Lee et al., SPELLING PROCESSES OF CHILDREN WITH NONSYNDROMIC CLEFTS

Although the incidence of CL/P is high among Asian children (Vanderas, 1987; Yi et al., 1999), most of the research to date has focused on children with CL/P living in North America or Europe. Recent work has investigated the cognitive and communicative skills of Asian children with CL/P (Fong, 2008; Lu et al., 2010; Young et al., 2010; Young et al., 2012), but it does not cover literacy skills. In contrast, there is an extensive body of knowledge on literacy development on typically developing (TD) children, and this provided a starting point for the present study (see Bishop and Snowling, 2004, on dyslexia and specific language impairment; Treiman and Bourassa, 2000, on spelling; Rayner et al., 2001, on reading; Rickard Liow and Tng, 2003, and Yeong and Rickard Liow, 2011, 2012, on spelling development and phonological awareness in Asian children). From this body of work, it is clear that phonological awareness and memory skills are both important for literacy development, and so there is a need to understand how children with CL/P compare with children without CL/P on these cognitive-linguistic measures. Cognition and Language Skills in Children With CL/P The past five decades of research on specific cognitive and language skills of children with CL/P has resulted in mixed findings. However, there is a clear trend of delayed development and underachievement compared with TD peers from infancy through to adolescence (Jocelyn et al., 1996; Speltz et al., 2000; Persson et al., 2012). Broder et al. (1998) and Buckenberger (1990) reported high learning disability rates of 46% and 51%, respectively, in school-aged children with CL/P. Interactions between cleft type and gender also revealed that boys with cleft palate only (CP) had an even higher learning disability rate of 79% (Broder et al., 1998). These rates contrast sharply with rates of between 10% and 20% in the general population (Rutter et al., 2004; Altarac and Saroha, 2007). Difficulties in multiple language domains, namely semantics, syntax, phonology, and pragmatics, have also been documented in the CL/P population (Nation, 1970; McWilliams et al., 1990; Broen et al., 1998; Frederickson et al., 2006; Kummer, 2008). Young et al. (2010) also estimated that bilingual Chinese preschoolers with CL/P are 3.9 to 12.7 times more likely than their peers without CL/P to have expressive language difficulties. Such problems appear to have a long-lasting impact. Poor academic achievement during secondary education was reported in a recent population-based study by Persson et al. (2012), where Swedish adolescents with CL/P were found to be more likely to obtain the lowest grades in English, mathematics, and Swedish and less likely to obtain high grade-point averages compared with TD peers. However, achievement seems to be related to cleft type. Compared with a rate of only 2.74% among TD

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adolescents, 7.10% of adolescents with cleft lip and palate (CLP) and 9.89% of adolescents with CP were not awarded secondary school graduation certificates; whereas, the rate for adolescents with cleft lip only (CL) was 2.52%, which was not significantly higher than that of TD adolescents. The impact of cleft type on cognitive and language performances is considered again later in this article. Explanations for the increased risk of cognitive, speech, and language difficulties in the CL/P population are wide ranging and inconclusive. Extrinsic factors (e.g., surgical timing, frequent hospitalization, lack of language stimulation) and intrinsic factors (e.g., abnormal dentition, velopharyngeal inadequacy, fluctuating conductive hearing loss) have been proposed (see Kuehn and Moller, 2000, for a review). Studies evaluating the neurobiology of learning patterns indicating differences ˇ in cortical auditory dysfunction (Ceponien_ e et al., 1999), neuropsychological functioning (Richman, 1995), brain development (Conrad et al., 2009; Weinberg et al., 2009), and neural activation patterns (Goldsberry et al., 2006) in individuals with clefts compared with healthy controls have also been reported, suggesting deviant neurological and neuroanatomical contributions to cognitive and language difficulties. These differences highlight the need for further investigations into spelling performances of children with CL/P. High rates of reading disability (between 30% and 40%) have also been observed in the non-Asian CL/P population (Richman et al., 1988; Broder et al., 1998) compared with rates of between 10% and 15% among the non-Asian general population (Lyon, 1999). Richman et al. (1988) reported an overall rate of 35% for moderate reading disability and 17% for severe reading disability in their group of 6- to 13-year-olds with CLP or CP. Age and cleft type effects were also identified. Reading disability rates were highest in 6- to 7-year-olds (48.6% in children with CLP and 53.5% in children with CP). However, in older children aged 10 to 13 years, rates decreased to 8.6% in children with CLP and 33.3% in children with CP. Subsequent work by Richman and colleagues (2003, 2005) further suggested that reading difficulties in children with CL/P are related to dysnomia (word-finding problems), and they recommended intervention focused on oral-based phonics to enhance phonological awareness rather than a sightword reading approach. In contrast, the rate and severity of literacy disorders in Asian children with CL/P are not well understood. Without more knowledge of the difficulties that Asian children with CL/P face in reading and spelling, the rationale for assessment practices is hard to justify, and planned interventions may be inadequate. Set against these concerns, a recent study by Collett et al. (2010a) found no differences between the performances of children with CL/P (CLP, n ¼ 29; CP, n ¼ 28)

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and 77 controls on receptive language, expressive language, and basic reading tasks at 5 and 7 years old. Nevertheless, Collett et al. (2010b) found that children with CL/P (CL, n ¼ 8; CLP, n ¼ 22; CP, n ¼ 12; 28 boys; mean age, 6 years 10 months) showed poorer performance on letter-word identification, reading fluency, passage comprehension, nonword repetition, and sightword and nonword reading compared with age-matched controls (n ¼ 43; 28 boys; mean age, 6 years 10 months). To summarize, the existing research shows that children with CL/P are at greater risk of language and literacy difficulties, leading to poorer outcomes in school. Thus far, there is one study suggesting that the spelling abilities of children with CL/P are in the normal range. Stackhouse (1982) compared the spelling performance of ten 6- to 11-year-old children with CL/P with that of age-matched controls on the Schonell Graded Word Spelling test (Schonell, 1965). She found no quantitative differences between the two groups, and the qualitative analyses of spelling responses revealed that children with CL/P were using phoneme-grapheme conversion rules. However, the sample size of 10 per group was small given the wide age range of the participants, and the graded nature of the test means that an average 6-year-old need only spell 10 simple words with a consonant-vowel-consonant (CVC) phonological structure (e.g., bed, ran, mat). Apart from this study 30 years ago on a limited set of words, there have been no investigations on the spelling abilities of children with CL/P. Phonological Awareness and Memory The importance of phonological awareness for literacy development is well documented in TD children (Caravolas et al., 2001; Ehri et al., 2001). Phonological awareness is defined as ‘‘a multilevel skill of breaking down words into smaller units’’ (Gillon, 2004, p. 4) and consists of three different but related levels. The first level, syllable awareness, refers to an understanding that a word can be divided into syllables. The second level, onset-rime awareness, is the understanding of how a syllable is composed of two units known as the onset and the rime. The last level, phoneme awareness, relates to an understanding of how a syllable can be divided into individual phonemes or sounds (Gillon, 2004). Each of these levels can be tested using a variety of tasks; for example, phoneme awareness can be tested using alliteration tasks, phoneme matching, phoneme isolation, phoneme deletion, and phoneme segmentation (refer to Gillon, 2004, for a list of tasks to examine an individual’s ability at each level). Phoneme awareness is consistently found to correlate strongly with spelling abilities (Stuart and Masterson, 1992; van Bon and Duighuisen, 1995; Gillon, 2004), and longitudinal studies on TD preschool and elementary

school children have provided support for this relationship (Wagner et al., 1993; Burgess and Lonigan, 1998; Lonigan et al., 1998; Wood and Terrell, 1998; Bowey, 2005). In their cross-linguistic longitudinal study, Furnes and Samuelsson (2011) also found that kindergarten children’s phonological awareness skills predicted spelling abilities at grade 1 school level across their American, Australian, and Scandinavian samples. Further evidence comes from intervention studies that document that phonemic awareness training with a focus on orthography results in better reading and writing performance (e.g., Gillon, 2005). Moreover, testing phonological processing skills with nonword tasks has proved particularly useful because the child is required to use letter-sound and sound-letter correspondences in order to decode and encode the novel strings. As yet, the relationship between phoneme awareness and spelling skills in the CL/P population remains unclear. Wilson (2007) and Collett et al. (2010b) examined both processes but neither study reported significant disparities in the phonological awareness abilities of 4- to 7-year-old children with CL/P and TD children. However, the results of the same study by Collett et al. (2010b) showed that children with CL/P performed significantly more poorly in nonword repetition compared with controls. Given that other work on nonword processing shows that nonword repetition may be a marker of reading and spelling impairments in TD children (Baird et al., 2011), this result suggests that phonological memory may be impaired in children with CL/P. Arguments relating to a unitary versus fractionated memory system supporting cognitive and language learning have resulted in a tripartite model of working memory (WM) being proposed by Baddeley and Hitch (1974), where WM is necessary for the concurrent storage and manipulation of information. Three components, each responsible for a different function, make up the WM model. The first component, the phonological loop, is responsible for storing and rehearsing verbal information and is thought to be necessary for the acquisition of language (i.e., verbal short-term memory [STM]). The second component in the WM model is the visuospatial sketchpad, which is responsible for storing and manipulating visual information (i.e., visuospatial STM). The last component, the central executive, is important for coordinating the processes in the phonological loop and the visuospatial sketchpad. These memory processes have been found to influence language learning and higher-order reasoning (Eliason and Richman, 1990; Montgomery, 2002; Gathercole et al., 2006). Related research on children with CL/P has generally indicated verbal memory functions to be within normal limits and visual memory abilities to be deficient. For example, Eliason and Richman (1990) reported average

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auditory memory spans with significant delays in visual and intersensory memory measures among 65 children 4 to 6 years old with CL/P. Similar findings were reported by Richman et al. (2005) when they studied the relationships between verbal and visuospatial STM and reading disorders in 48 children 7 to 9 years old with CL/P. They found better performance in verbal STM compared with visuospatial STM, with 65% of the children with CL/P having significant visuospatial STM deficits that correlated significantly with poor reading scores. In an effort to profile the memory abilities of children with CL/P in relation to their reported higher prevalence of language difficulties, Young et al. (2012) compared the performances of 86 Mandarin-English speaking bilingual children with CL/P (CL, n ¼ 15; unilateral CLP, n ¼ 19; bilateral CLP, n ¼ 10; CP, n ¼ 12; 41 boys; mean age, 5 years 7 months) with 100 Mandarin-English speaking bilingual TD children (60 boys; mean age, 4 years 11 months) in receptive vocabularies as well as verbal and visuospatial STM and WM, using language dominance and the presence of cleft as grouping variables. Using subtests from the Automated Working Memory Assessment (Alloway, 2007), they found that regardless of the presence of cleft, English-dominant children performed significantly better than Mandarindominant children in the processing component of the Listening Recall subtest (verbal WM). In contrast to the findings by Collett et al. (2010b), there were no differences between the CL/P and TD groups in terms of English and Mandarin receptive vocabulary scores or for nonword recall. On the other hand, Conrad et al. (2009) found poorer performance in 66 children with CL/P (CL, n ¼ 14; CLP, n ¼ 30; CP, n ¼ 22; 42 boys; mean age, 12 years 7 months) compared with 87 agematched controls (43 boys; mean age, 12 years 6 months) on a range of verbal neuropsychological tasks (rapid verbal labeling, verbal fluency, verbal memory). These mixed findings warrant further work, and thus far, no researchers have specifically looked at how verbal STM and WM may be related to spelling in children with CL/P.

difficulties compared with children with CL or unilateral or bilateral CLP (e.g., Broen et al., 1998; Priester and Goorhuis-Brouwer, 2008; Young et al., 2010). Vallino, Zuker, and Napoli (2008) reported rates of 13% for speech impairments and 18% for language impairments in 95 children with CL (68 boys; mean age, 3 years 1 month). These rates are much higher than the rates of between 5.6% and 8% reported in the general population (Law et al., 2000). Conrad et al. (2009) found that across cleft types (CLP, CL, CP), there were no differences in performance or verbal IQ, but children with CP had significantly lower verbal IQ than performance IQ; whereas, children with CL had lower performance IQ than verbal IQ. Clearly, the relationships among cleft types, cognition, and language performance are complex and require further clarification. For this preliminary study on spelling, the relatively small sample size meant that the relationship between cleft type and spelling could not be considered. To summarize, the results of empirical work on the cognitive-linguistic and phonological precursors of literacy development in children with CL/P remain inconclusive. More research is needed because these skills are strongly related to academic performance (Dockrell, 2001; Scarborough, 2001; Lewis et al., 2006), and children with CL/P are reported to be underachievers (Broder et al., 1998; Persson et al., 2010). Given that spelling is both speech based (Jalil and Rickard Liow, 2008) and cognitively demanding compared with reading (Bosman and Van Orden, 1997), spelling performance could provide new insights on any atypical underlying processing, such as deficits in phonological awareness and/or verbal memory in children with CL/P. The aims of this preliminary study were twofold: (1) to test the hypothesis that children with CL/P will perform more poorly than TD children on verbal STM, verbal WM, phonological awareness, and word and nonword spelling measures; and (2) to examine the degree to which verbal memory and phonological awareness scores are correlated with performance on word and nonword spelling.

Language and Cognitive Differences Related to Cleft Type

Ethics approval for this study was granted by the SingHealth Centralised Institutional Review Board. Parental consent and participant assent were obtained for all participants.

Mixed findings have also been reported regarding the development of cognition, language, and academic functions related to cleft type. Higher rates of learning disability have been reported in children with CP than in children with CLP (Broder et al., 1998), and some studies have suggested that children with CP have more pervasive language and cognitive difficulties contributing to poor learning (e.g., Lamb et al., 1973; Richman and Eliason, 1986). Other studies suggested that children with CP have lower rates of language

METHOD

Participants CL/P Group. A total of 15 children (nine boys) in the CL/P group were recruited from a hospital-based multidisciplinary center in Singapore dedicated to cleft and craniofacial care. The following inclusion criteria were used: (1) diagnosis of any form of nonsyndromic CL/P; (2) aged between 6 years 0 month and 8 years 11

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months; (3) primary repairs for CL/P performed within the first year of life as part of the routine surgical protocol; and (4) English as their dominant language. The surgical team performed lip repair and/or rhinoplasty at 3 months of age and palate repair at 9 months of age. Language dominance was determined based on parental report. Children with medical diagnoses of other syndromes, congenital disorders, developmental delays, and hearing impairments were excluded from the study. The final CL/P group consisted of six children with bilateral CLP, three children with unilateral CLP, one child with bilateral CL, one child with unilateral CL, two children with posterior CP, and two children with submucous cleft palate. Their mean age was 7 years 6 months. TD Group. A total of 15 age- and sex-matched TD children were recruited from kindergartens and afterschool care centers. The same exclusion criteria applied to the TD group. None of the TD children had any identified medical conditions, hearing, physical, cognitive, or learning impairments. All the TD children had English as their dominant language based on parental report. Their mean age was 7 years 4 months. An independent t test confirmed no significant difference in the mean age of children between groups, t(28) ¼ 0.35, P . .05. Procedures All participants completed a battery of hearing, speech, verbal memory, phonological awareness, and spelling assessments, in that order and within 90 minutes. Test stimuli for the verbal memory, word, and nonword spelling tasks were recorded by a native English-Mandarin female speaker and presented to participants through free-field speakers. Participants’ responses to the speech, verbal memory, and phonological awareness tasks were audio- and video-recorded for rescoring purposes. All children in the CL/P group were assessed in a quiet therapy room at the hospital-based center; whereas, all children in the TD group were assessed in a quiet room at their schools or homes. Before the assessment began, participants were told that they could request a break whenever they were tired. Measures Hearing Screening. All participants underwent a puretone audiometry screening using a Madsen Micromate 304 Portable Screening Audiometer (GN Otometrics, Taastrup, Denmark). Participants failed the hearing screen if their pure-tone average at 1000, 2000, and 4000 Hz exceeded 20 dB hearing loss (HL) for one or both ears (American Speech-Language-Hearing Association, 1997). The criterion of 20 dB HL was used to identify participants at risk of minimal hearing loss (MHL), and

those who failed the screening at this level were referred to the otolaryngologist for further evaluation (American Academy of Audiology, 2011). Another criterion of 25 dB HL was adopted to determine exclusion from the study because this screening level was reported to have the best combined sensitivity and specificity rates for educationally significant hearing loss (ESHL) compared with a level of 20 dB HL (American Academy of Audiology, 2011). Participants who obtained scores above 25 dB HL were excluded from the study and subsequent data analysis. Speech Evaluation. The Articulation subtest of the Diagnostic Evaluation of Articulation and Phonology (DEAP; Dodd et al., 2002) was used to evaluate the presence of articulatory disorders and to provide qualitative information about each participant’s speech production. This was necessary because the phonological awareness tasks required the participants to give responses orally. Participants were therefore not penalized for articulatory errors in their responses during the phonological awareness tasks. Participants were asked to name 30 target pictures of CVC phonological structure (e.g., ‘‘pig’’). All responses were phonetically transcribed by the coinvestigator (a speech-language pathologist with 6 months of experience in craniofacial speech pathology and cleft speech transcription). Phonological Awareness. Two phoneme awareness measures, with differing task complexity, were devised to assess the participants’ phonological awareness skills at the phoneme level (see 1). The first task was a 15-item phoneme isolation task that required the participants to verbally identify the target sound (e.g., ‘‘What is the last sound in dog?’’). The second, a 15-item phoneme deletion task, was chosen as a more demanding task because it required the participants to identify and delete a target sound and verbally blend remaining sounds together (e.g., ‘‘Say the word dog without the ‘g’ sound.’’; Yopp, 1988). Verbal Memory. Two tasks from the Working Memory Test Battery for Children (WMTB-C; Pickering and Gathercole, 2001) were used to evaluate verbal STM and verbal WM, respectively. Verbal STM was operationalized using digit recall, where the participant was asked to listen to a sequence of numbers and then repeat it. Verbal WM was operationalized using backward digit recall. The participant was asked to listen to a sequence of numbers and repeat the sequence backward. The test stimuli for both tasks were presented at a speech rate of one digit per second. There were two practice trials for each verbal memory task. Participants progressed to the next block if there were four correct responses in a block, each block consisting of six trials. Testing was discontinued when participants made three errors in the same block. Word Spelling. This was assessed using the Green list from the Wide Range Achievement Test–4th Edition

Lee et al., SPELLING PROCESSES OF CHILDREN WITH NONSYNDROMIC CLEFTS

with 11 years of experience in craniofacial speech pathology and cleft speech transcription and who is a native speaker of Singapore English) randomly rescore 10% of all the recorded data on the articulation, verbal memory, phonological awareness, and spelling tasks. The percentages of agreement were 95% for the DEAP articulation task and 100% for the digit recall, backward digit recall, phoneme isolation, phoneme deletion, word spelling, and nonword spelling tasks. Data Analysis. Parametric tests were unsuitable for analyzing digit recall, phoneme isolation, word spelling approximation, and nonword spelling due to the heterogeneity of variance. To investigate whether the performance of children with CL/P was different from that of TD children, the data were analyzed in two ways: (1) nonparametric Wilcoxon signed-rank test to compare the performance of both groups on verbal memory, phonological awareness, and word and nonword spelling tasks; and (2) nonparametric Spearman correlation analyses to explore the relationships between each of the verbal memory and phonological awareness task with word and nonword spelling, respectively, for each group. RESULTS Hearing and Speech Measures Hearing Screening. Based on the screening level of 20 dB HL for MHL, all participants passed the screening audiometry except for one child with CL/P and two TD children. The child with CL/P failed the screening on the left ear with an average score of 25 dB HL. One TD child failed the screening on the left ear with an average score of 25 dB HL; whereas, the other TD child failed the screening on the right ear with an average score of 22 dB HL. However, based on the screening level of 25 dB HL for ESHL, all participants in the CL/P and TD groups passed the hearing screening and data from all participants were included in subsequent data analysis. Speech. The speech responses of each participant were analyzed qualitatively. Of the TD children, 86% (13 children) had age-appropriate speech patterns. One TD child presented with lateralized /s/ productions; whereas, another TD child presented with interdentalized /s, z/ productions. Of the TD children, 73% (11 children) deleted final / /, and 40% of them stopped /h, ð/ to /t, d/. Of those with CL/P, 67% presented with age-appropriate speech patterns and normal resonance. Two children with CL/P displayed cleft-type speech characteristics of backing, active nasal fricatives, nasal air emission, and nasal turbulence. Two other children with CL/P presented with lateralized or interdentalized /S, Z, S, tS/. One child with CL/P presented with a delayed phonological pattern of stopping for /v/. Of the children with CL/P, 33% deleted final / /; whereas, 40% of them stopped /h, ð/ to /t, d/. The r

r

(WRAT-4; Wilkinson and Robertson, 2006), a U.S.normed test designed to assess basic academic skills in reading, spelling, and math computation for individuals aged 5 to 94 years. The word spelling test was administered according to the WRAT-4 manual instructions. Participants were asked to spell their name (2 points), 13 English letters (13 points), and 42 target words (42 points). The target words were presented twice—singly and in a disambiguating sentence—and scored for accuracy and approximation. Spelling accuracy was scored such that a correct spelling for a target word (e.g., ‘‘dress’’) was awarded 1 point and an incorrect spelling (e.g., ‘‘dreas’’) was not awarded a point. Spelling approximation was scored using the selforganizing lexical acquisition and recognition algorithm from MatchCalc (see Davis, 1999, for an overview), a program developed for measuring similarities between letter strings and a target word, such that ‘‘dress’’ received 1 point and ‘‘dreas’’ received 0.86 points. Using MatchCalc, the target word and the participant’s response are entered into the program. MatchCalc returns a point between 0.0 and 1.0 that is based on how orthographically similar the participant’s response is compared with the target word. Nonword Spelling. Nonword spelling is an especially sensitive, nonlexical measure of a child’s phonological processing abilities using sound-letter rules to produce a phonologically plausible response, and this contrasts with word spelling, where a child might have a representation in the orthographic lexicon (Kohnen et al., 2009). This was assessed via a 20-item nonword list devised by the research investigators (see Appendix 2), with due regard to the possible sound combinations allowed in English (Rastle et al., 2002). This nonword test targeted phonemes (plosives, fricatives, and affricates) that are prone to being distorted in the speech of children with CL/P. Stimuli were presented twice, with two practice trials before the test items. Phonologically plausible responses for each item were awarded 1 point (e.g., ‘‘kipa’’/‘‘keepa’’). Intrarater Reliability. Ten percent of all the recorded data on the articulation, verbal memory, phonological awareness, and spelling tasks for each experimental group was randomly selected. Data were rescored by the coinvestigator. The percentage of agreement was calculated by dividing the number of agreements by the total number of agreements and disagreements. The percentages of agreement were 97% for the DEAP articulation task and 100% for the digit recall, backward digit recall, phoneme isolation, phoneme deletion, word spelling, and nonword spelling tasks. The high levels of agreement is similar to findings alluded to in Cordes (1994), who cites more than 80% agreement as moderate to excellent reliability. Interrater Reliability. This was determined by having the principal investigator (a speech-language pathologist

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TABLE 1 Summary Statistics on Measures of Verbal Memory, Phonological Awareness, and Word and Nonword Spelling CL/P Group

TD Group

Tasks (Maximum Score)

Mean

SD

Mean

SD

P

Digit recall (54) Backward digit recall (36) Phoneme isolation (15) Phoneme deletion (15) Word spelling total (57) Word spelling accuracy (42) Word spelling approximation (42) Nonword spelling (20)

29.47 13.07 13.6 11.47 29.6 14.73 28.61 3.73

7.02 4.48 1.24 2.59 6.5 6.33 8.04 3.01

29.93 13.67 13.93 13.73 30.2 15.13 31.37 6.93

3.96 5.25 0.8 1.03 5.09 5.1 3.86 2.15

.33 .73 .289 .011* .95 .975 .211 .017*

† CL/P ¼ children with cleft lip and/or palate, TD ¼ typically developing children, ns ¼ nonsignificant. * P , .05 (two-tailed).

two processes of final / / deletion and stopping of /h, ð/ are common phonetic features of English spoken in Singapore (Deterding, 2007). r

Cognitive-Linguistic Measures Phonological Awareness. The CL/P group’s performance (median ¼ 11) was significantly poorer than that of the TD group (median ¼ 14) on the phoneme deletion task, T ¼ 6.5, P , .05, r ¼ .47. Spelling Nonword Spelling. The CL/P group’s performance (median ¼ 3) was significantly poorer than that of the TD group (median ¼ 8), T ¼ 8.5, P , .05, r ¼ .44. No other significant differences were found (Table 1). Correlational Relationships Between Variables Because ceiling effects were observed across both CL/ P and TD groups for the name and letter spelling components in the WRAT-4 spelling task, only the word spelling scores were used for the correlation analyses. Table 2 shows the one-tailed Spearman correlation statistic for the CL/P and TD groups. For the TD group, the correlations between (1) verbal STM and nonword spelling, (2) verbal WM and word spelling, and (3) phonological awareness (both phoneme isolation and phoneme deletion) and word spelling were

positive and moderate to strong (Field, 2009; all P , .05). Verbal WM (backward digit recall) showed the strongest association with word spelling approximation for the TD group (rs ¼ .69, P , .01). For the CL/P group, the correlations between (1) verbal STM and word spelling, (2) verbal STM and nonword spelling, (3) phoneme deletion and word spelling, and (4) phoneme deletion and nonword spelling were positive and moderate to strong. In contrast to the TD group, phonological awareness (phoneme deletion) showed the strongest association with word spelling approximation for the CL/P group (rs ¼ .73, P ¼ .001). DISCUSSION Previous work shows that children with CL/P often underachieve on education (Persson et al., 2010) and have a relatively weak foundation in literacy. These problems appear to be the result of deficits in their cognitive-linguistic abilities. The twofold aim of this preliminary study was (1) to test the hypothesis that children with CL/P will perform more poorly than TD children on verbal STM, verbal WM, phonological awareness, and word and nonword spelling measures; and (2) to examine the degree to which verbal memory and phonological awareness scores are correlated with performance on word and nonword spelling. The sample size was small, but the findings from this study on an Asian population provide evidence that children with CL/P perform more poorly in phoneme deletion and nonword spelling compared with TD children. Nonword spelling ability gauges the child’s ability to use speech-based phonological knowledge to spell novel stimuli independently. The data suggest that children with CL/P are less able to spell sequences of phonemes they have not seen in print. Moreover, the relationships between the underlying processes for spelling appear to be different for children with CL/P and TD children. The next section discusses the outcome for each of the cognitive-linguistic tasks administered and considers the theoretical and practical implications. Verbal Memory Previous work (Conrad et al., 2009; Young et al., 2010) suggested that children with CL/P have deficits in

TABLE 2 Correlation Coefficients Between Verbal Memory, Phonological Awareness, and Spelling Measures for Children With Cleft Lip and/or Palate (CL/P) and Typically Developing (TD) Children Word Spelling Accuracy

Word Spelling Approximation

Nonword Spelling

Cognitive-Linguistic Measures

CL/P

TD

zdiff†

CL/P

TD

zdiff

CL/P

TD

zdiff

Digit recall Backward digit recall Phoneme isolation Phoneme deletion

.57* .22 .15 .70**

.08 .63** .45* .57*

1.78* 1.27 .82 .54

.66** .27 .14 .73***

.19 .69** .39 .56*

1.47 1.40 .66 .72

.61** .34 .01 .70**

.49* .11 .24 .12

.42 .60 .58 1.82*

† zdiff ¼ significance of difference between correlation coefficients. * P , .05 (one-tailed), ** P , .01 (one-tailed), *** P , .001 (one-tailed);

Lee et al., SPELLING PROCESSES OF CHILDREN WITH NONSYNDROMIC CLEFTS

learning, but our finding suggests that young children with CL/P embark on literacy tasks with similar verbal memory capacities as those of TD children. Data obtained in the present study suggest that verbal memory in children with CL/P may be a relative strength. This lends support to earlier findings by Eliason and Richman (1990) and Richman et al. (2005) that stimuli presented verbally to children with CL/P were recalled better than stimuli presented visually. However, WM capacities develop and increase between 4 and 14 years of age (Alloway et al., 2006); therefore, any potential differences between the CL/P and TD groups in this study might not yet be apparent. A second explanation is that verbal STM and WM were tested using numbers as the stimuli. The familiarity of the stimuli may have minimized any potential differences between the groups because numeracy is one of the early skills taught to children in local kindergartens (Brebner et al., 1996). This prior knowledge of numbers may explain the longer memory span demonstrated (Hulme and Maughan, 1991) because more cognitive resources can be allocated to process and rehearse the sequential presentation of the digits. Future work could include memory tasks with unfamiliar stimuli such as nonword repetition. Phonological Awareness Despite the use of different tests, the results of the present study supported the work of Wilson (2007) and Collett et al. (2010b) that the phoneme isolation ability (sometimes referred to as sound segmentation skill) of children with CL/P is not significantly different from that of TD children. However, given that a range of phoneme awareness skills is crucial for spelling success (Gillon, 2004; Furnes and Samuelsson, 2011), the CL/P group’s poorer performance on phoneme deletion, a more demanding phonological task than phoneme isolation, suggests that they could be disadvantaged in their independent acquisition of orthographic knowledge for novel words. This could have the effect of limiting self-teaching (i.e., the independent use of phonological knowledge to decode and recode words), such that consolidation of important orthographic knowledge is delayed (see Shahar-Yames and Share, 2008). This kind of disadvantage could deepen when target words become increasingly complex in phonological structure and could require the child to use more advanced phonological skills in order to map out possible orthographic representations. In the studies by Collett and colleagues (2010a, 2010b) and Wilson (2007), phoneme deletion was not included as part of the assessment. Given the differences found in the present study, a thorough examination of different levels of phonological awareness (syllable, onset-rime, and phoneme levels) and more demanding phoneme

77

awareness tasks are recommended for clinical assessments and future research work. Despite the CL/P group’s comparable strength in phoneme isolation (Collett et al., 2010b; Wilson, 2007), this skill is probably not sufficient in supporting the CL/P group in nonword spelling. Intervention studies have shown that phonological awareness training helps to improve reading and spelling in TD children (Gillon, 2005), but providing equivalent training for children with CL/P may not be effective if their underlying phonological representations are different or less well established than those of TD children. In other words, it might be important to establish strong phonological representations for speech and naming before embarking on the training of higher-order phonological awareness skills for print that is often implemented for TD children’s literacy development. Therefore, before phonological remediation can be offered to children with CL/P, more work is needed to clarify the relationships among phonological awareness, reading, and spelling in children with CL/P. Word Spelling The ceiling effects on name and letter spelling components for both the CL/P and TD groups may be explained by the demands of the local curriculum. Children will have been drilled in alphabet recitation, number and letter recognition, and writing practice during their preschool years (Gupta et al., 1998). Weinrich and Fay (2007) have also suggested that the development of phonological awareness requires both letter knowledge and phoneme-grapheme correspondence skills. Letter knowledge precedes and contributes to the acquisition of phoneme-grapheme correspondence rules (Whitehurst and Lonigan, 1998; Roth and Baden, 2001). Of note, the ceiling effects on letter spelling in this study suggest that the children already possess basic knowledge of the alphabet as a foundation for literacy development and that this component of most literacy assessments is overlearned and therefore not sufficient for differentiating the good from the poor spellers. Given the CL/P group’s significantly weaker performance on the phoneme deletion task, we expected poorer word spelling performance because the children would need to make use of phonological skills when attempting to recode increasingly complex target words. The most likely explanation for the observed lack of a difference is that young children with CL/P are able to keep pace by rote-learning words in the early stages of spelling. The high-frequency items in the WRAT-4 Green list (Wilkinson and Robertson, 2006) could number among the words in the sight-word vocabulary being taught at the preschool level (e.g., boy, cat, dress). The positive relationship found between verbal STM

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and word spelling in children with CL/P supports this explanation. Previous exposure or additional support from parents and teachers of children with CL/P involving orthographic information of the word or letter sequences that do not require detailed phonological support could have contributed to better early spelling performance. As with letter spelling, assessment of word spelling is less likely to be as useful as nonword spelling for identifying those at risk of weak, independent spelling. Not surprisingly, phonological awareness was significantly related to word spelling for both CL/P and TD groups, supporting previous research that phonological skills underlie spelling development (Caravolas et al., 2001; Furnes and Samuelsson, 2011). What is interesting is that despite having poorer phonological processing abilities compared with the TD group, the CL/P group performed as well in word spelling, further supporting our reasoning that they may be using nonphonological or sequencing strategies to spell words. More evidence could be seen when the performance of the CL/P group in nonword spelling was analyzed. Nonword Spelling The CL/P group’s significantly poorer nonword spelling performance was expected given their poorer phonological awareness abilities, because unlike word spelling, they cannot rely on previous rote learning or the lexical route. Spelling nonwords forces a child to use the nonlexical route, in which the CL/P group is weaker and, therefore, less successful than their TD peers. However, the correlational analysis revealed that nonword spelling correlated positively with verbal STM but not with any measure of phonological awareness for TD children, indicating that the ability to spell nonwords might be related to how well verbal information is being stored and processed in conjunction with good phonological awareness skills. Despite phoneme deletion being positively correlated with nonword spelling for the children in the CL/P group, their poorer nonword spelling performance indicated that their ability to manipulate complex phonological skills might still be inadequate. Planning spelling intervention for children with CL/P will require the integration of multiple modalities so that they learn various strategies to aid in recalling spelling. Shahar-Yames and Share (2008) suggested that spelling novel words instead of only reading them allows for more effective and consistent learning outcomes in spelling because the process of spelling allows for the self-teaching, or independent acquisition, of orthographic, phonological, visuospatial, and motor-kinesthetic information. Their views are supported by Bosman and Van Orden (1997), who recommended that strategies to encourage correct spelling need to take

into the account the relationships between phonology, orthography, and semantics. Limitations The design of the present study covered a range of skills related to memory (verbal STM and WM only) and phonological awareness (phoneme-level tasks; i.e., phoneme isolation and phoneme deletion). A major limitation of this study was the small sample size, resulting in the need to consider the theoretical and practical implications of the results with care. Nevertheless there is good reason to think that the spelling and writing skills of children with CL/P develop differently from those of TD children. Another consequence of the limited sample size was that each cleft subgroup was extremely small. As a consequence, it was not possible to examine sex or cleft-type differences. Although early identification of spelling problems is important, it seems likely that larger discrepancies between groups of CL/P and TD children would be observed with age. This is because the children with CL/ P do not appear to have developed a foundation for independent spelling, judging from their poorer phoneme deletion and nonword spelling performance, but at 7 years of age their rote learning is sufficient to keep pace. A follow-up study with a sample of older participants would be informative. Future Directions Our understanding of the cognitive-linguistic underpinnings of literacy development in children with CL/P is still extremely limited. Future research investigating literacy development on a larger CL/P sample should explore (1) verbal and nonverbal memory profiles; (2) phonological awareness profiles; and (3) reading and writing abilities. Recruiting a larger CL/P sample will be beneficial for exploring differences in performances according to sex and cleft type. Future work could also investigate the role of multilingualism on reading and spelling development, especially in the Singaporean context, where children are typically exposed to a second language other than English (e.g., Mandarin, Malay, or Tamil). CONCLUSIONS This is the first spelling study in recent years that compared Asian children with CL/P with matched controls on experimental tasks. The findings suggest that children with CL/P performed less well in phoneme deletion and nonword spelling tasks compared with TD children. The relationships between verbal memory and phonological skills with word and nonword spelling were also found to be different between children with CL/P and TD children. Clinically, the findings from this study suggest the

Lee et al., SPELLING PROCESSES OF CHILDREN WITH NONSYNDROMIC CLEFTS

importance of speech-language pathologists including phonological awareness (especially phoneme deletion) and spelling assessments (words and nonwords) as part of their management protocols with children with CL/P in order to identify children in need of early intervention. This is pertinent to the Asian context where the rate of clefting is one of the highest in the world, indicating that a larger number of children with CL/P may be at risk for poor literacy development. Acknowledgments. We thank all the children who participated in the study, their parents, and the principals of the participating schools for their assistance and support. We also thank Genevieve Ng, Melanie Chastan, and Vincent Yeow for their assistance with various aspects of the study.

REFERENCES Alloway, TP. Automated Working Memory Assessment. London: Pearson Education; 2007. Alloway TP, Gathercole SE, Pickering SJ. Verbal and visuospatial short-term and working memory in children: are they separable? Child Dev. 2006;77:1698–1716. Altarac M, Saroha E. Lifetime prevalence of learning disability among U.S. children. Pediatrics. 2007;119:S77–S83. American Academy of Audiology. Childhood hearing screening guidelines. Available at http://www.audiology.org/resources/ documentlibrary/Documents/ChildhoodScreeningGuidelines.pdf. Accessed October 10, 2011. American Speech-Language-Hearing Association. Guidelines for audiologic screening. Available at http://www.asha.org/docs/html/ GL1997-00199.html. Accessed October 10, 2011. Baddeley AD, Hitch GJL. Working memory. In: Bower GA, ed. The Psychology of Learning and Motivation: Advances in Research and Theory. New York: Academic Press; 1974:47–89. Baird G, Slonims V, Simonoff E, Dworzynski K. Impairment in nonword repetition: a marker for language impairment or reading impairment? Dev Med Child Neurol. 2011;55:711–716. Bishop D, Snowling M. Developmental dyslexia and specific language impairment: same or different? Psychol Bull. 2004;130:858–886. Bosman AMT, Van Orden GC. Why spelling is more difficult than reading. In: Perfetti CA, Rieben L, Fayol M, eds. Learning to Spell: Research, Theory, and Practice Across Languages. Hillsdale, NJ: Lawrence Erlbaum Associates; 1997:173–194. Bowey JA. Grammatical sensitivity: its origins and potential contribution to early work reading skill. J Exp Child Psychol. 2005;90:318–343. Brebner C, Gupta AF, Wong A. The Singapore Pro-Ed Chart. Singapore 1996. Adapted with permission from Gard A, Gilman L, Gorman J. Speech & Language Development Chart, 2nd ed. Austin, TX: Pro-Ed; 1993. Broder H, Richman L, Matheson P. Learning disability, school achievement, and grade retention among children with cleft: a twocenter study. Cleft Palate Craniofac J. 1998;35:127–131. Broen P, Devers M, Doyle S, Prouty J, Moller K. Acquisition of linguistic and cognitive skills by children with cleft palate. J Speech Lang Hear Res. 1998;41:676–687. Buckenberger LM. Learning Disabilities in the Cleft Lip and Palate Population. Chicago: DePaul University; 1990. Dissertation. Burgess SR, Lonigan CJ. Bidirectional relations of phonological sensitivity and prereading abilities: evidence from a preschool sample. J Exp Child Psychol. 1998;70:117–141. Caravolas M, Hulme C, Snowling MJ. The foundations of spelling ability: evidence from a 3-year longitudinal study. J Mem Lang. 2001;45:751–774.

79

ˇ Ceponien_ e R, Hukki J, Cheour M, Haapanen ML, Ranta R, Naatanen R. Cortical auditory dysfunction in children with oral clefts: relation with cleft type. Clin Neurophysiol. 1999;110:1921– 1926. Collett BR, Leroux B, Speltz ML. Language and early reading among children with orofacial clefts. Cleft Palate Craniofac J. 2010a;47:284–292. Collett BR, Stott-Miller M, Kapp-Simon KA, Cunningham ML, Speltz ML. Reading in children with orofacial clefts versus controls. J Pediatr Psychol. 2010b;35:199–208. Conrad AL, Richman L, Nopoulos P, Dailey S. Neuropsychological functioning in children with non-syndromic cleft of the lip and/or palate. Child Neuropsychol. 2009;15:471–484. Cordes AK. The reliability of observational data: I. Theories and methods for speech-language pathology. J Speech Hear Res. 1994;37:264–278. Davis C. The Self-Organizing Lexical Acquisition and Recognition (SOLAR) Model of Visual Word Recognition. Sydney: University of New South Wales; 1999. Dissertation. Deterding D. Singapore English. Edinburgh: Edinburgh University Press; 2007. Dockrell JE. Assessing language skills in preschool children. Child Psychol Psychiatry Rev. 2001;6:74–85. Dodd B, Hua Z, Crosbie S, Holm A, Ozanne A. Diagnostic Evaluation of Articulation and Phonology. London: Harcourt Assessment; 2002. Ehri LC, Nunes SR, Willows DM, Schuster BV, Yaghoub-Zadeh Z, Shanahan T. Phonemic awareness instruction helps children learn to read: evidence from the National Reading Panel’s meta-analysis. Read Res Q. 2001;36:250–287. Eliason M, Richman LC. Language development in preschoolers with cleft. Dev Neuropsychol. 1990;6:173–182. Field AP. Discovering Statistics Using SPSS. 3rd ed. London: Sage Publications; 2009. Fong YT. The Language Abilities of Cantonese-Speaking Children With Cleft Palate: A Pilot Study. Hong Kong: The University of Hong Kong; 2008. Dissertation. Frederickson M, Chapman K, Hardin-Jones M. Conversational skills of children with cleft lip and palate: a replication and extension. Cleft Palate Craniofac J. 2006;43:179–188. Furnes B, Samuelsson S. Phonological awareness and rapid automatized naming predicting early development in reading and spelling: results from a cross-linguistic longitudinal study. Learn Individ Differ. 2011;21:85–95. Gathercole SE, Alloway TP, Willis CS, Adams AM. Working memory in children with reading disabilities. J Exp Child Psychol. 2006;93:265–281. Gillon GT. Facilitating phoneme awareness development in 3- and 4year-old children with speech impairment. Lang Speech Hear Serv Sch. 2005;36:308–324. Gillon GT. Phonological Awareness: From Research to Practice. New York: Guilford Press; 2004. Goldsberry G, O’Leary D, Hichwa R, Nopoulos P. Functional abnormalities in the neural circuitry of reading in men with nonsyndromic clefts of the lip or palate. Cleft Palate Craniofac J. 2006;43:683–690. Grunwell P, Russell J. Phonological development in children with cleft lip and palate. Clin Linguist Phon. 1988;2:75–95. Gupta AF, Brebner C, Chandler Yeo H. Developmental assessments in speech-language therapy in Singapore. Asia Pac J Speech Lang Hear. 1998;3:17–28. Harding A, Grunwell P. Characteristics of cleft palate speech. Int J Lang Commun Disord. 1996;31:331–357. Hulme C, Maughan S. Memory for familiar and unfamiliar words: evidence for a long-term memory contribution to short-term memory span. J Mem Lang. 1991;30:685–701.

80

Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1

Jalil S, Rickard Liow SJ. How does home language influence early spellings? Phonologically plausible errors of diglossic Malay children. App Psycholinguist. 2008;29:535–552. Jocelyn LJ, Penko MA, Rode HL. Cognition, communication, and hearing in young children with cleft lip and palate and in control children: a longitudinal study. Pediatrics. 1996;97:529–534. Kohnen S, Nickels L, Castles A. Assessing spelling skills and strategies: a critique of available resources. Aust J Learn Difficulties. 2009;14:113–150. Kuehn D, Moller K. Speech and language issues in the cleft palate population: the state of art. Cleft Palate Craniofac J. 2000;348:1– 348:35. Kummer AW. Cleft Palate and Craniofacial Anomalies: Effects on Speech and Resonance. 2nd ed. Clifton Park, NY: Delmar Cengage Learning; 2008. Lamb M, Wilson F, Leeper H. The intellectual functions of cleft palate children compared on the basis of cleft type and sex. Cleft Palate J. 1973;10:367–377. Law J, Boyle J, Harris F, Harkness A, Nye C. Prevalence and natural history of primary speech and language delay: findings from a systematic review of the literature. Int J Lang Commun Disord. 2000;35:165–188. Lewis BA, Shriberg LD, Freebairn LA, Hansen AJ, Stein CM, Taylor HG, Iyengar SK. The genetic bases of speech sound disorders: evidence from spoken and written language. J Speech Lang Hear Res. 2006;49:1294–1312. Lonigan CJ, Burgess SR, Anthony JL, Barker TA. Development of phonological sensitivity in 2- to 5-year-old children. J Educ Psychol. 1998;90:294–311. Lu Z, Ma L, Luo Y, Fletcher P. The effects of unrepaired cleft palate on early language development in Chinese infants. Cleft Palate Craniofac J. 2010;47:400–404. Lyon RG. The NICHD Research Program in Reading Development, Reading Disorders and Reading Instruction. Bethesda, MD: National Center for Learning Disabilities; 1999. McCutchen D, Teske P, Bankston C. Writing and cognition: implications of the cognitive architecture for learning to write and writing to learn. In: Bazerman C, ed. Handbook of Research on Writing. Mahwah, NJ: Erlbaum; 2008:447–465. McWilliams BJ, Morris HL, Shelton RL. Language disorders. In: McWilliams BJ, Morris HL, Shelton RL, eds. Cleft Palate Speech. 2nd ed. Philadelphia: BC Decker; 1990:236–246. Montgomery JW. Understanding the language difficulties of children with specific language impairments: does verbal working memory matter? Am J Speech Lang Pathol. 2002;11:77–91. Morris H, Ozanne A. Phonetic, phonological, and language skills of children with a cleft palate. Cleft Palate Craniofac J. 2003;40:460– 470. Nation JE. Vocabulary comprehension and usage of preschool cleft palate and normal children. Cleft Palate J. 1970;7:639–644. Persson M, Becker M, Svensson H. Academic achievement in individuals with cleft—a population-based register study. Cleft Palate Craniofac J. 2012;49:153–159. Peterson-Falzone SJ, Hardin-Jones MA, Karnell MP. Cleft Palate Speech. 3rd ed. St. Louis: Mosby; 2001. Pickering S, Gathercole S. Working Memory Test Battery for Children (WMTB-C). London: Psychological Corporation; 2001. Priester GH, Goorhuis-Brouwer SM. Speech and language development in toddlers with and without cleft palate. Int J Pediatr Otorhinolaryngol. 2008;72:801–806. Rastle K, Harrington J, Coltheart M. 358,534 nonwords: the ARC nonword database. Q J Exp Psychol. 2002;55A:1339–1362. Rayner K, Foorman BR, Perfetti CA, Pesetsky D, Seidenberg M. How psychological science informs the teaching of reading. Psychol Sci Public Interest. 2001;2:31–74. Richman LC. Neuropsychological development in adolescents: cognitive and emotional model for considering risk factors for adolescents with cleft. Cleft Palate Craniofac J. 1995;32:99–103.

Richman LC, Eliason M. Development in children with cleft lip and/or palate: intellectual, cognitive, personality, and parental factors. Semin Speech Lang. 1986;7:225–239. Richman LC, Eliason MJ. Disorders of communication: developmental language disorders and the cleft palate. In: Walker CE, Roberts MC, eds. Handbook of Clinical Child Psychology. 3rd ed. New York: John Wiley & Sons; 2001:603–620. Richman LC, Eliason MJ, Lindgren SD. Reading disability in children with clefts. Cleft Palate J. 1988;25:21–25. Richman LC, Ryan SM. Do the reading disabilities of children with cleft fit into current models of developmental dyslexia? Cleft Palate Craniofac J. 2003;40:154–157. Richman LC, Wilgenbusch T, Hall T. Spontaneous verbal labeling: visual memory and reading ability in children with cleft. Cleft Palate Craniofac J. 2005;40:565–569. Rickard Liow SJ, Tng SK. Biscriptal literacy development of Chinese children in Singapore. In: McBride-Chang C, Chen HC, eds. Reading Development in Chinese Children. Westport, CT: Praeger; 2003:215–228. Roth FP, Baden B. Investing in emergent literacy intervention: a key role for speech-language pathologists. Semin Speech Lang. 2001;22:163–173. Rutter M, Caspi A, Fergusson D, Horwood LJ, Goodman R, Maughan B, Moffitt TE, Meltzer H, Carroll J. Sex differences in developmental reading disability: new findings from 4 epidemiological studies. JAMA. 2004;291:2007–2012. Scarborough HS. Connecting early language and literacy to later reading (dis)abilities: evidence, theory, and practice. In: Neuman SB, Dickinson DK, eds. Handbook of Early Literacy Research. New York: Guilford Press; 2001:97–110. Schonell FJ. Schonell Spelling Test. Edinburgh: Oliver & Boyd; 1965. Shahar-Yames D, Share DL. Spelling as a self-teaching mechanism in orthographic learning. J Res Read. 2008;31:22–39. Speltz ML, Endriga MC, Hill S, Maris CL, Jones K, Omnell ML. Brief report: cognitive and psychomotor development of infants with orofacial clefts. J Pediatr Psychol. 2000;25:185–190. Stackhouse J. Reading and spelling in speech disordered children. Br J Disord Commun. 1982;17:53–60. Stuart M, Masterson J. Patterns of reading and spelling in 10-year-old children related to prereading phonological abilities. J Exp Child Psychol. 1992;54:168–187. Treiman R. The foundations of literacy. Curr Dir Psychol Sci. 2000;9:89–92. Treiman R, Bourassa D. The development of spelling skills. Top Lang Disord. 2000;20:1–18. Treiman R, Goswami U, Tincoff R, Leevers H. Effects of dialect on American and British children’s spelling. Child Dev. 1997;68:229– 245. Vallino LD, Zuker R, Napoli JA. A study of speech, language, hearing, and dentition in children with cleft lip only. Cleft Palate Craniofac J. 2008;45:485–494. van Bon WHJ, Duighuisen HCM. Sometimes spelling is easier than phonemic segmentation. Scand J Psychol. 1995;36:82–94. Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: a review. Cleft Palate J. 1987;24:216–225. Wagner RK, Torgesen JK, Laughon P, Simmons K, Rashotte CA. Development of young readers’ phonological processing abilities. J Educ Psychol. 1993;85:83–103. Weinberg SM, Andreasen NC, Nopoulos P. Three-dimensional morphometric analysis of brain shape in nonsyndromic orofacial clefting. J Anat. 2009;214:926–936. Weinrich B, Fay E. Phonological awareness/literacy predictors of spelling abilities for first-grade children. Contemp Issues Commun Sci Disord. 2007;34:94–100. Whitehurst GJ, Lonigan CJ. Child development and emergent literacy. Child Dev. 1998;69:848–872. Wilkinson GS, Robertson GJ. Wide Range Achievement Test 4 (WRAT-4). Lutz, FL: Psychological Assessment Resources; 2006.

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Wilson K. Phonological Awareness, Speech, and Language Skills in Children With Clefts. Chapel Hill: University of North Carolina; 2007. Dissertation. Wood C, Terrell C. Preschool phonological awareness and subsequent literacy development. Educ Psychol. 1998;18:253–274. Yeong SHM, Rickard Liow SJ. Cognitive-linguistic foundations of early spelling development in bilinguals. J Educ Psychol. 2011;103:470–488. Yeong SHM, Rickard Liow SJ. Development of phonological awareness in English-Mandarin bilinguals: a comparison of English-L1 and Mandarin-L1 kindergarten children. J Exp Child Psychol. 2012;112:111–126.

APPENDIX 1

Yi NN, Yeow VKL, Lee ST. Epidemiology of cleft lip and palate in Singapore—a 10-year hospital-based study. Ann Acad Med Singapore. 1999;28:655–659. Yopp HK. The validity and reliability of phonemic awareness tests. Read Res Q. 1988;23:159–177. 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;74:456–464. Young SE, Purcell AA, Ballard KJ, Rickard Liow SJ, Da Silva Ramos S, Heard R. Bilingual children with nonsyndromic cleft lip and/or palate: language and memory skills. J Speech Lang Hear Res. 2012;55:1314–1328.

Phonological Awareness Tasks Phoneme Isolation

1 2 3 4 5 6 7 8 9 10 11 12 13 14 15

APPENDIX 2 Trial 1 Trial 2 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20

81

Cap Pen Cup Mess Meal Cash Tall Book Go Dog Fish Sing Zoo Love Van

What What What What What What What What What What What What What What What

is is is is is is is is is is is is is is is

the last sound in cap? the last sound in pen? the first sound in cup? the last sound in mess? the first sound in meal? the last sound in cash? the first sound in tall? the first sound in book? the first sound in go? first sound in dog? the first sound in fish? the last sound in sing? the first sound in zoo? the first sound in love? the first sound in van?

Nonword Spelling Task Zil Nop Po Ib Chof Zogi Bluch Nisp Kipa Glub Smung Tarf Lodden Brast Napish Krads Ush Ballop Zorp Joo Dakes Swont

Phoneme Deletion Say Say Say Say Say Say Say Say Say Say Say Say Say Say Say

cap without the /p/ sound. pen without the /n/ sound. cup without the /k/ sound. mess without the /s/ sound. meal without the /m/ R sound. cash without the / / sound. tall without the /t/ sound. book without the /b/ sound. go without the /g/ sound. dog without the /d/ sound. fish without the /f/ sound. sing without the /n/ sound. zoo without the /z/ sound. love without the /l/ sound. van without the /v/ sound.