FAQs from Cleft Palate and Craniofacial Committee | IALP : International Association of Communication Sciences and Disorders (IALP) FAQs from Cleft Palate and Craniofacial Committee – IALP : International Association of Communication Sciences and Disorders (IALP)

FAQs from Cleft Palate and Craniofacial Committee

Introduction

A craniofacial disorder is an umbrella term that refers to a structural abnormality of the skull and/or the face. Most craniofacial disorders are present at birth, and the type and severity of the abnormality can vary widely. The most common craniofacial disorder is Cleft Palate + Lip (see below). Less common and rare craniofacial disorders include craniofacial dysostoses syndromes such as Apert syndrome and Crouzon syndrome, which are  characterised by premature closure of fibrous joints (cranial sutures) between various skull bones (craniosynostosis), as well as distinctive facial abnormalities (e.g. reduction in growth of the upper jaw, cheekbones and eye sockets). Craniosynostosis results in an abnormally shaped skull which can restrict growth of the brain that can lead to increased pressure in the brain requiring medical intervention. Other types of craniofacial disorders include microtia (malformation of the outer ear) and hemifacial microsomia (underdevelopment of one or both sides of the face).

The rest of this document focuses on Cleft Palate + Lip,  within a context where a Cleft Lip and Palate Team and/or speech and language therapy (SLT) services are available. Where there is limited or no access to  SLT services there have been initiatives to use other health care staff to provide advice and intervention (Sell et al., 2008). These include training other health care professionals to deliver speech and language therapy (D’Antonio 2003), speech camps where intensive therapy is delivered by a trained SLT while simultaneously training other health care professionals (Wirt et al, 1990 a and b, Pamplona et al 2004, 2005) and training community based workers (Balasubramaniyan et al., 2018).

 

What is Cleft Palate +/- Lip?

Cleft Palate + Lip (CP+L) is the most common congenital craniofacial disorder. It can manifest in various expressions, ranging from a small split of the upper lip (i.e. incomplete cleft lip) or the soft palate (i.e. bifid uvula) to a cleft of the upper lip, alveolus, and the soft and hard palate (i.e. complete cleft lip and palate). Moreover, the cleft can occur on one side of the face (i.e. unilateral) or both sides of the face (i.e. bilateral).

Cleft Palate + Lip is the result of disrupted embryological development of the primary palate (upper lip and alveolus) and secondary palate (hard palate and velum) during the first trimester of pregnancy. The disruption to the formation of the lip and palate is caused by genetic or a chromosomal condition, or environmental factors (e.g. cigarette smoke, alcohol, maternal use of several medicinal drugs etc.). Although the exact causal factors remain unknown, consensus exists about the multifactorial nature of the causes (Kummer, 2020).

Orofacial clefts arise in 1.7 per 1000 live born babies (Mossey & Castilla, 2003); however, the prevalence varies when variables such as cleft type, gender and ethnicity are considered. Regarding cleft type, cleft lip with or without cleft palate is more prevalent compared to cleft palate only (0.91 vs. 0.62 per 1000) and cleft lip and palate arises more often in comparison with cleft lip only (Calzolari et al., 2004, 2007). A predominance in females is observed for cleft palate (male/female-ratio 0.83 to 0.93), whereas cleft lip with or without cleft palate most typically occurs in males (male/female-ratio 1.70 to 1.81) (Mossey et al., 2009; Calzolari et al., 2004, 2007; Mossey & Castilla, 2003). Studies of ethnic and geographical differences in prevalence of orofacial clefts, showed overall higher prevalence of cleft lip and/or palate in Asians, a lower prevalence for Africans and an intermediate prevalence for Caucasians. This racial and ethnical variation might be explained by a mixture of differences in environmental risk factors and genetic predisposition.

 

What are the potential consequences of Cleft Palate + Lip??

Feeding: In the first year of life, feeding may be a challenge if there is a palate involvement. Feeding difficulties in CP+L centre on the infant’s inability to suck, which causes a disruption of the oral phase of swallowing (Miller, 2011). The infant is unable to create negative intraoral pressure due to the unrepaired cleft palate which prevents a separation of the oral from the nasal cavities. This can result in increased feeding times, nasal regurgitation, inadequate volume of oral intake, excessive intake of air, feeding fatigue and the possible consequential ulceration of the nasal mucosa (Bannister, 2001; Miller, 2011), all of which may impact negatively on weight gain. See guidelines below on feeding.

Middle ear diseases: Middle ear diseases such as otitis media with effusion and acute otitis media frequently occur in young children with CP+L up to 5-6 years of age (Goudy et al., 2006). The high incidence  of middle ear problems in CP+L is often due to Eustachian tube dysfunction as a result of aberrations of the tensor veli palatini muscle where the functions of the Eustachian tube are to provide aeration of the middle ear, equalization of pressure between the middle ear and atmospheric pressure, and drainage of any middle ear fluid and secretions into the nasopharynx (Petersone-Falzone, Hardin-Jones & Karnell, 2010), although the exact relationship between the tensor veli palatini muscle and Eustachian tube function in CP+L is still not well-defined (Heidsieck, Smarius & Oomen, 2016). Additionally, children with CLP show deviations in the anatomy of the Eustachian tube cartilages as well as abnormal insertion of the tensor veli palatini and levator veli palatini muscles, often leading to malfunctioning (Lennox, 2001) resulting in accumulation of fluid in the middle ear  which may cause a conductive hearing loss varying from 10 to 40 dB (Lennox, 2001). Such a hearing loss can have a significant negative effect on the development of speech and language (Willging & Kummer, 2008).

Dental abnormalities: Dental care begins in infancy. The early stages of dental development are characterised by delayed eruption of primary teeth on the cleft side, congenital absence of primary teeth, abnormal morphology of teeth, supernumerary (extra teeth) around the cleft site, resulting in an increased risk of dental carries and hence, a focus on oral hygiene (Huebener, 2016). Dental development continues to be characterised by dental anomalies and many dental interventions are required at the mixed dentition stage between the ages of 6 to 12 years of age (Huebener, 2016). Repair of the lip and palate can also result in narrowing of the (palate) arch form resulting in anterior and posterior crossbites (Daskalogiannakis & Antonarakis, 2016) potentially affecting oral hygiene and speech articulation.

Facial growth: Abnormal or deficient facial growth is also a potential consequence in CP+L, attributable to a combination of intrinsic developmental deficiencies, iatrogenic as a result of treatment and functional or adaptive factors (Kreiborg et al., 2013). Abnormal facial growth becomes increasingly evident during the pubertal growth spurt resulting in a concave facial profile and a class III jaw relationship (Ross, 1987; Semb, 1991) corresponding with the time when appearance becomes an important element for self-esteem and sexual identity (Bradbury, 2005). A reported 10-50% of individuals with CP+L will require orthognathic surgery to correct this facial deformity (e.g. Good, Mulliken & Padwa, 2007; Friede, Liljia & Lohmander, 2011), which is typically undertaken around 17-18 years of age, when active facial growth is complete. The surgery can have a negative impact on speech and velopharyngeal function, necessitating pre- and post-operative assessments (e.g. Pereira, Sell & Tuomainen, 2013).

Speech difficulties: Speech difficulties in CP+L range from early speech difficulties to speech difficulties related to velopharyngeal insufficiency and/or speech difficulties related to dental anomalies and class III occlusal status. These are described in detail in a later section.

Language and educational achievement: Large-scale population studies have shown evidence of poorer academic achievement resulting in non-attainment of school leaving certificate (Persson et al., 2012) and an increased rate of requiring a statement of educational needs, or extra help in school (Fitzsimons et al., 2018). Children with CP+L may also have difficulties with reading (e.g. Conrad, Richman & Nopoulus, 2015), and mathematics, attributable to deficits in rapid naming and visual-spatial skills (Goodwin et al., 2017).  In terms of language, toddlers with CP+L show language delay compared to their normal aged peers, however, these differences tend to narrow with increasing age (Lancaster et al., 2019). Although published studies show no statistically significant differences at the school-age level, the child with CP+L may still be facing language difficulties, performing below the mean or at the lower end of the normal range (e.g. Klinto et al., 2015; Boyce et al., 2018). These persistent language difficulties may be an underlying cause of difficulties with reading and mathematics (Paul, Norbury & Gosse, 2018) and thus warrants monitoring and/or intervention where appropriate.

Psychosocial aspects and (health-related) quality of life: Potential risks associated with CP+L are medical risks associated with having a birth defect (e.g. feeding in infancy), functional and/or cognitive problems (e.g. speech and language, academic achievement) and psychological stress (e.g. stresses on the family around treatment options and medical interventions) (Kapp-Simon & Gaither, 2016), which can impact on the individual’s social and emotional adjustment and well-being, self-concept, self-esteem, satisfaction with appearance, body image and quality of life (Hunt et al., 2005; Kapp-Simon & Gaither, 2016; Stock & Feragen, 2016). In spite of these many challenges faced by the individual with CP+L across the lifespan, reviews of studies have concluded that the impact of having a CP+L on psychological aspects and quality of life is low (Hunt et al., 2005; Stock & Feragen, 2016) and that many individuals lead ‘happy and productive lives’ (Kapp-Simon & Gaither, 2016; p. 397). For example, the review by Stock & Feragen (2016) concluded that although dissatisfaction with appearance may be a specific concern in CP+L, this is comparable to or better than levels reported in the general population, and that there is no strong evidence that anxiety and depression levels are raised in CP+L, although this may be raised in social situations. As psychological adjustment can fluctuate across the lifespan, formal psychological support should be given even in childhood and continued on into adulthood (Kappen et al., 2019). Resistance factors such as family strengths and adaptation (e.g. family cohesion and emotional well-being, social/financial support) and the child’s resilience (e.g. hardiness, spirit) are necessary elements and early identification and management of potential problems are crucial (Kapp-Simon & Gaither, 2016).

 

What are typical speech characteristics in patients with Cleft Palate + Lip?

Normal velopharyngeal closure is the coordinated action of the velum (soft palate), the lateral pharyngeal walls and the posterior pharyngeal walls (Kummer 2008). Velopharyngeal dysfunction (VPD) in speech is a generic term used to describe impairment of function due to structural deficits (velopharyngeal insufficiency), neurological disorders (velopharyngeal incompetency) and faulty learning (velopharyngeal mislearning) (D’Antonio & Scherer 1995). Atypical articulatory errors often referred to as cleft speech characteristics (CSCs) are associated with cleft palate and/or velopharyngeal dysfunction (VPD) (Sell et al., 1999; Peterson-Falzone et al., 2006). Cleft speech consists not only of consonant sound errors but abnormal resonance (hypernasality, hyponasality) and nasal airflow errors (audible nasal emission, nasal turbulence) may also be present.

Articulatory errors can be due to structural aetiologies such as a fistula or velopharyngeal insufficiency (described as passive characteristics) or due to active mislearning sometimes associated with a history of VPD, and frequently these co-occur. CSCs tend to be errors in place of articulation, where the sound is produced further back in the oral cavity e.g.alveolar targets are produced as palatals or velars/uvulars or further back in the larynx, pharynx, or velopharynx as glottal stops, pharyngeal fricatives or active nasal fricatives. Passive errors may affect manner of articulation resulting in weak, nasalised or absent oral pressure consonants. Children with cleft palate may also have phonological delay and/or disorder.

Articulatory errors may also be attributable to a class III jaw relationship which tends to affect production of speech sounds such as labiodental fricatives e.g. /f/ (which may be produced as reverse labiodentals) and alveolars /t/ and /s/ (which may be dentalised or interdentalized) (Vallino, 1990; Pereira et al., 2008). These articulation errors are not responsive to speech therapy and tend to improve usually spontaneously following orthognathic surgery though not necessarily to normal status (Lee et al., 2002; Pereira, 2012).

The most frequently occurring resonance disorder is hypernasality. Hypernasality refers to excess nasal resonance perceived during speech production due to a coupling of the oral and nasal resonating cavities (Sweeney, 2011). Nasal emission and nasal turbulence comprise the escape of audible air through the nasal cavity during the production of oral pressure consonants (plosives, fricatives and affricates). In the case of nasal emission, the sound quality is frictional but not turbulent or snorting (Sweeney, 2011). It can be due to incomplete closure of the velopharyngeal sphincter or a palatal fistula. Nasal turbulence is distinguished from nasal emission as the sound quality is turbulent or snorting and it is often due to a small gap in the velopharyngeal sphincter. Both nasal emission and nasal turbulence accompany the oral consonant, unlike the nasal fricative CSC which replaces the consonant.

Early speech and language development may be compromised in children with CP+L. Young children may experience delay relative to their non-affected peers across multiple speech and language constructs, performing significantly below their peers on measures of consonant inventory, speech accuracy, expressive language and receptive language (Lancaster et al. 2019). Infants may have a later onset of babbling, which can be characterised by fewer canonical syllables and fewer oral consonants compared with non-cleft infants. Instead, they produce more nasal consonants, more posterior sounds than anterior sounds eg pharyngeal and glottal realizations both in babble and early word productions across languages (Chapman et al., 2011; Chapman & Willadsen, 2011; Willadsen, 2007). They have smaller consonant inventories, both in terms of the types of sounds (plosives, fricatives), individual sounds and less complex syllable and word structures (Chapman & Willadsen, 2011).

Delay in language may be evident especially expressive language in children with non-syndromic CP+L (Hardin-Jones & Chapman, 2014; Morgan et al., 2017; Richman, 1980; Scherer, Boyce, et al., 2013; Watkins et al., 2018; Young et al., 2010). Differences between affected children and their typically developing peers appear to decrease with age (Lancaster et al., 2019).

 

How are speech disorders in patients with Cleft Palate + Lip assessed and diagnosed?

Speech and language disorders in patients with CP+L will be assessed and diagnosed by the speech and language therapist (SLT). In some areas, two SLTs may be involved with the child, one in the community and one specialist SLT working with the cleft and craniofacial team. During the first two years, assessment often includes checklists such as the Receptive-Expressive Emergent Language Scale (REEL-3) (Bzoch et al., 2006) that uses reports by parents/carers to identify receptive and expressive language problems. The SLT may listen to and analyse a sample of babble or early vocalisations either in the clinic or recorded at home by parents.

Articulation can be assessed at word level using a standardized picture-naming test and detailed phonetic transcription. Pictures can be used to elicit words which contain all single speech sounds in different  word positions and various consonant clusters. At sentence level, patients can be asked to read or repeat short sentences loaded with high pressure consonants (plosives, fricatives and affricates). The target sounds should be phonetically transcribed.  Following this, errors should be categorized according to their Cleft Speech Characteristics. Stimulability is an important diagnostic aspect of assessment. Stimulability refers to the elicitation of a target sound or an approximate of the target sounds using auditory, visual and tactile cues. This indicates the child’s potential for speech improvement with therapy and also indicates which sounds should be targeted in therapy. Speech intelligibility is usually assessed by the SLT using perceptual ratings of speech understandability or by parent’s perception of their child’s understandability using a scale such as the Intelligibility in Context Scale (McLeod et al., 2012)

Assessment of resonance and nasal airflow errors is usually performed by perceptual evaluation of connected speech, automatic speech (i.e. counting, days of the week) and repetition of sentences using a 4- or 5-point rating scale (e.g. normal, borderline, mild, moderate, severe). When assessing nasal airflow errors it is important to distinguish between nasal emission/turbulence which accompanies sound production and nasal fricatives, articulatory errors which replace oral consonants with a devoiced nasal fricative sound.

Instrumental measurements can be used to confirm the presence of hypernasality. This can be performed using the Nasometer (Kay Elemetrics Corporation, 2010). The Nasometer is a device that contains a sound separator plate, which is placed on the upper lip. The microphones in front of the nose and the mouth collects all the nasal and oral acoustic energy, respectively. These signals are sent to a computer that calculates the nasalance score. Nasalance values can vary considerably across different languages and regional dialects and hence the assessor should be aware of normative nasalance values for the child’s language/dialect when interpreting results.  Instrumental assessment should always be used in conjunction with perceptual results.

If perceptual assessments indicate possible velopharyngeal dysfunction, a differential diagnosis can be made using direct visualization of the velopharyngeal sphincter. The two most common methods are videofluoroscopy and nasendoscopy which provide information which can be video recorded about the structure, movement and closure/non closure of the velopharyngeal sphincter (Sell & Pereira, 2011). Multiview videofluoroscopy involves low dose continuous x-ray of the velopharyngeal sphincter during speech using different views of the sphincter. Nasendoscopy involves passing a flexible fibre-optic scope through the nose as far as the nasopharynx in order to observe the soft palate, the lateral and posterior pharyngeal walls and surrounding structures (Sell & Pereira, 2011). Results of these assessments should be reviewed by the cleft team and secondary speech surgery may be recommended for hypernasality, nasal airflow errors and passive cleft speech characteristics such as weak intraoral pressure and nasal realisations of consonants.

 

How can speech disorders in patients with Cleft Palate + Lip be treated?

All children with CP+L should be seen by an SLT of the cleft team within the first year. They may be referred to local SLT services for assessment and management of communication development around 9-12 month or older depending on the service. Children can be regularly monitored and if a problem with communication (speech or language) is identified, parents should be given advice on how to help stimulate normal speech and language development.  Early intervention for speech and language problems is advised in infants known to be at risk, as is the case with cleft palate. When children with cleft palate present with any of the following: delay in receptive language, delay in use of words and gestures, reduced vocalisations, a lack of plosive sounds and canonical babble by 11 months, early intervention is recommended (Hardin-Jones & Chapman, 2019). Early intervention approaches for children with CP+L can be delivered by the SLT  or often it can focus on parent training/education, with the emphasis on speech sound development, phonological interventions and increase in vocabulary, using programmes that simultaneously intervene in speech and language areas (Scherer & Louw, 2011). Naturalistic approaches such as Enhanced Milieu Teaching and focused stimulation have been found to improve sound production and vocabulary in children with cleft palate (Scherer et al., 2008; Kaiser et al., 2017). In young children, speech may be targeted using indirect therapy such as Multi Sensory Input Modelling (MSIM) (Harding & Bryan, 2000) and MSIM with Output (MSIM+O) (Calladine, 2009), where a high dose of models of target speech sounds are provided to the child with the aim of creating new speech motor programs (Calladine & Vance, 2019).

As the child gets older, around 2½ or 3 years of age, they may be able to cooperate with direct therapy. If they present with language problems these should be addressed in therapy along with speech errors. For speech difficulties, therapy should focus on errors of articulation as hypernasality and accompanying nasal emission/turbulence are usually associated with a structural problem. Direct therapy is usually initiated when the child can cooperate for a short session and is stimulable on sound production, however, it may take several sessions to elicit a sound. Two main approaches to therapy include the traditional articulation intervention approach (Van Riper & Erickson, 1996) and a phonological approach, such as minimal pair therapy (Barlow & Geirut, 2002) or multiple opposition intervention (WIlliams, 2000; Boers 2015). In the articulation approach, speech therapy will primarily focus on the correction of articulation errors by teaching the correct place and manner of articulation for all affected consonants. In order to achieve these objectives, different specific therapy techniques can be used (Kummer, 2011, Peterson Falzone et al., 2006). Using a phonological approach, therapy targets phonological patterns rather than individual sounds (Harding-Bell and Howard, 2011). Often a combined phonological and articulatory approach to therapy is successful (Pamplona et al., 1999; Sweeney et al., 2020).

When velopharyngeal insufficiency has been diagnosed by the cleft team, secondary surgical management of the palate and/or velopharyngeal sphincter is required. Such secondary speech-improving surgery can only resolve speech disorders that are directly caused by the abnormal structure or function of the cleft (e.g. hypernasality, nasal emission, weak production of plosives or fricatives) and active errors or disorders caused by mislearning will not  spontaneously improve (Kummer, 2011).

Finally, a prosthetic approach can sometimes be considered as well. Indications to initiate a prosthetic treatment are absence of the soft palate or a large anatomic defect and failed primary surgical treatment (Sell et al., 2006). Furthermore, prosthetic treatment can be used to evaluate the potential advantages of surgical treatment or as a temporary solution to facilitate the treatment of compensatory articulation errors by the speech-language therapist.

 

How can babies with unrepaired Cleft Palate + Lip be supported during feeding?

In order to facilitate the feeding process, oral feeding facilitation techniques as well as specialized feeding equipment are often recommended (Miller, 2011; Cleft Palate Foundation, 2009). Below are some common methods that can help with oral feeding:

  1. Positioning the infant in an upright position of at least 60 degrees. This will allow gravity to assist with posterior transfer of fluid and with swallowing. In addition, it will help prevent nasal regurgitation (i.e. liquids or food passing through the nose during drinking or eating).
  2. Oral-motor techniques such as stabilization of the jaw or closing the lip with the thumb. These techniques can be used to facilitate the nutritive intake.
  3. Assisted feeding. This can be used to help the child compensate for the inability to create a negative pressure in the mouth. As such, the feeder will squeeze the nipple in synchrony with the infant’s sucking pattern.
  4. Specialised teats and bottles. Specialized feeding equipment can be used to assist feeding in patients with cleft lip and/or palate. A wide variety of specialized nipples, bottles and cups is available. Overall, a nipple with a broad base is preferred in patients with a cleft lip as this may assist closure of the lip during feeding. Moreover, the size of the nipple hole needs to be adjusted depending on the infant’s drinking speed. In addition, some nipples are designed to release milk by the infant’s lip pressure instead of by sucking or the nipples can be squeezed by the feeder. Similarly, special bottles and cups are designed to avoid the need for active infant sucking during feeding.

Oral feeding facilitation techniques or feeding equipment are determined for each individual child by the nurse in consultation with the parents. Once the lip and palate are closed, the feeding problems will generally disappear.

Where can patients with Cleft Palate + Lip find treatment?

Given the various interrelated problems associated with CP+L, a multidisciplinary team care approach is recommended, which typically includes a plastic and reconstructive surgeon, an orthodontist, an otorhinolaryngologist, a speech-language therapist, a maxillofacial surgeon, a social worker, a geneticist, a dentist, a psychologist, an audiologist and a paediatrician. Some cleft teams may also include a geneticist. Each specialist makes evaluations within their own discipline as well as interact closely with the other disciplines within the cleft team, in order to determine the appropriate timing and sequence of treatment, to optimize the functional, aesthetical and psychosocial outcomes, and to improve the patient’s quality of life.

 

REFERENCES

Introduction

Balasubramaniyan, S., Raghunathan, V., Rajashekhar, B., Sathiyasekaran, B. W. C., & Nagarajan, R. (2017). Planning community-based intervention for speech for children with cleft lip and palate from rural South India: A needs assessment. Indian Journal of Plastic Surgery, 50(03), 295-301

D’Antonio, L. L., & Nagarajan, R. (2003). Use of a consensus building approach to plan speech services for children with cleft palate in India. Folia phoniatrica et logopaedica, 55(6), 306-313.

Pamplona, M. C., Ysunza, A., & Ramı́rez, P. (2004). Naturalistic intervention in cleft palate children. International journal of pediatric otorhinolaryngology, 68(1), 75-81.

 

Pamplona, C., Ysunza, A., Patiño, C., Ramírez, E., Drucker, M., & Mazón, J. J. (2005). Speech summer camp for treating articulation disorders in cleft palate patients. International Journal of Pediatric Otorhinolaryngology, 69(3), 351-359.

 

Wirt, A., Wyatt, R., Sell, D. A., Grunwell, P., & Mars, M. (1990). Training assistants in cleft palate speech therapy in the developing world: a report. Cleft Palate Journal, 27(2), 169-174.

 

What is Cleft Lip and Palate?

Calzolari, E., Bianchi, F., Rubini, M., Ritvanen, A., Neville, A. J., & EUROCAT Working Group. (2004). Epidemiology of cleft palate in Europe: Implications for genetic research. Cleft Palate-Craniofacial Journal, 41(3), 244-249.

Calzolari, E., Pierini, A., Astolfi, G., Bianchi, F., Neville, A. J., & Rivieri, F. (2007). Associated anomalies in multi-malformed infants with cleft lip and palate: An epidemiologic study of nearly 6 million births in 23 EUROCAT registries. American Journal of Medical Genetics. Part A, 143A(6), 528-537.

Kummer, A. W. (2020). Cleft palate and craniofacial conditions: A comprehension guide to clinical management (4th ed.). Burlington, MA: Jones & Barlett Learning.

Mossey, P., & Castilla, E. (2003). Global registry and database on craniofacial anomalies: Report of a WHO Registry Meeting on Craniofacial Anomalies. Geneva, Switzerland: World Health Organization.

Mossey, P. A., Little, J., Muger R. G., Dixon, M. J., & Saw, W. C. (2009). Cleft lip and palate. The Lancet, 374(9703), 1773-1785.

What are the potential consequences in Cleft Lip and Palate?

Bannister, P. (2001) Early feeding management. In: Watson, A.C.H., Sell, D. and Grunwell, P. (Eds.) Management of cleft lip and palate. London, England, Whurr Publishers, 137-147.

Bradbury, E. & Bannister, P. (2005). Prenatal, perinatal and postnatal counselling. In A. C. H. Watson, D. Sell, & P. Grunwell (Eds.), Management of cleft lip and palate (pp. 117-122). London and Philadelphia: Whurr Publishers.

Boyce, J. O., Kilpatrick, N., Reilly, S., Da Costa, A., & Morgan, A. T. (2018). Receptive and expressive language characteristics of school-aged children with non-syndromic cleft lip and/or palate. International Journal of Language & Communication Disorders, 53(5), 959-968. doi:10.1111/1460-6984.12406.

Conrad, A. L., Richman, L., & Nopoulos, P. (2015). Reading achievement in boys with non-syndromic cleft palate only: Relationship to neuropsychological skill and neurocircuitry. Developmental Neuropsychology, 40(7-8), 395-406. doi:10.1080/87565641.2016.1142991.

Daskalogiannakis, J., & Antonarakis, G.S. (2016). Facial growth and development in individuals with Cleft. . In: J.E. Losee & R.E. Kirschner (Eds.), Comprehensive Cleft care. NY: Thieme, 1171-1195.

Friede, H., Lilja, J., & Lohmander, A. (2011). Long-term, longitudinal follow-up of individuals with UCLP after the Gothenburg primary early veloplasty and delayed hard palate closure protocol: maxillofacial growth outcome. The Cleft Palate-Craniofacial Journal. Advance online publication. doi: http://dx.doi.org/10.1597/10-252.

Good, P. M., Mulliken, J. B., & Padwa, B. L. (2007). Frequency of Le Fort I osteotomy after repaired cleft lip and palate or cleft palate. The Cleft Palate-Craniofacial Journal, 44, 396-401.

Goudy, S., Lott, D., Canady, J., & Smith, R. J. (2006) Conductive hearing loss and otopathology in cleft palate patients. Otolaryngology – Head and Neck Surgery, 134(6), 946-948.

Fitzsimons, K. J., Copley, L. P., Setakis, E., Charman, S. C., Deacon, S. A., Dearden, L., & van der Meulen, J. H. (2018). Early academic achievement in children with isolated clefts: a population-based study in England. Archives of Disease in Childhood, 103(4), 356-362. doi:10.1136/archdischild-2017-313777.

Hunt et al. Hunt, O., Burden, D., Hepper, P., & Johnston, C. (2005) The psychosocial effects of cleft lip and palate: a systematic review. European Journal of Orthodontics, 27(3), 274-285.

Huebener, D.V. (2016). Pediatric dentistry for the Cleft-affected patient. In: J.E. Losee & R.E. Kirschner (Eds.), Comprehensive Cleft care. NY: Thieme, 1197-1212.

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Kapp-Simon, K.A., & Gaither, R. (2016). Psychological and behavioural aspects of orofacial Clefting. In: J.E. Losee & R.E. Kirschner (Eds.), Comprehensive Cleft care. NY: Thieme, 383-401.

Klinto, K., Salameh, E. K., & Lohmander, A. (2015). Verbal competence in narrative retelling in 5-year-olds with unilateral cleft lip and palate. International Journal of Language & Communication Disorders, 50(1), 119-128. doi:10.1111/1460-6984.12127.

Kreiborg, S., Hermann, N. V., & Darvann, T. A. (2013) Characteristics of facial morphology and growth in infants with clefts. In: S. Berkowitz (Ed.), Cleft lip and palate: diagnosis and management. Miami, USA: Springer, 245-258.

Lancaster, H. S., Lien, K. M., Chow, J. C., Frey, J. R., Scherer, N. J., & Kaiser, A. P. (2019). Early speech and language development in children with nonsyndromic cleft lip and/or palate: a meta-analysis. Journal of Speech, Language, and Hearing Research 63(1), 14-31. doi:10.1044/2019_JSLHR-19-00162.

Miller, C.K. (2011). Feeding issues and interventions in infants and children with clefts and craniofacial syndromes. Seminars in Speech and Language, 32(2): 115-126.

Paul, R., Norbury, C. & Gosse, C. (2017). Language disorders from infancy through adolescence: Listening, speaking, reading, writing, and communicating (5th edition). St. Louis: Elsevier Inc.

Pereira, V.J., Sell, D., & Tuomainen, J. (2013). Effect of maxillary osteotomy on speech in cleft lip and palate: perceptual outcomes of velopharyngeal function. International Journal of Language and Communication Disorders, 48, 640-650.

Peterson-Falzone, S. J., Hardin-Jones, M., & Karnell, M. P. (2001). Cleft Palate speech. St. Louis, USA: Mosby.

Ross, R. B. (1987). Treatment variables affecting facial growth in complete unilateral cleft lip and palate. The Cleft Palate journal, 24, 5-77.

Semb, G. (1991). A study of facial growth in patients with bilateral cleft lip and palate treated by the Oslo CLP Team. Cleft Palate-Craniofacial Journal, 28, 22-39.

Stock, N.M., & Feragen, K.B. (2016). Psychological adjustment to cleft lip and/or palate: A narrative review of the literature, Psychology & Health, 31(7), 777-813.

What are typical speech characteristics in patients with cleft lip and/or palate?

How are speech disorders in patients with cleft lip and/or palate assessed and diagnosed?

How can speech disorders in patients with cleft lip and palate be treated?

Boers, M.,  Søgård Andersen, H., Dahl Jørgense, L.,  Willadsen, E. (2015).  Multiple Oppositions as an effective Tool to eliminate Compensatory Articulation in Children with Cleft Palate. Paper presented at 10th European Craniofacial Congress, Goteborg, Sweden.

Boyce, J. O., Kilpatrick, N., Reilly, S., Da Costa, A., & Morgan, A. T. (2018). Receptive and expressive language characteristics of school-aged children with non-syndromic cleft lip and/or palate. International Journal of Language & Communication Disorders, 53(5), 959-968. doi:10.1111/1460-6984.12406.

Bzoch, K.R., League, R. and Brown, V.L., 2003. Receptive-expressive Emergent Language Test: Examiner’s Manual. Pro-ed.

Calladine, S. (2009). Multi-sensory input modelling therapy intervention for young children with cleft palate. (Unpublished master’s thesis). Univ. of Sheffield, UK.

Calladine, S. & Vance, M. (2019). A psycholinguistic approach to therapy with very young children born with cleft palate. In A. Harding-Bell (Ed.), Case studies in cleft palate speech: Data analysis and principled intervention (pp.329-360). UK: J&R Press.

Chapman K. (2011). The relationship between early reading skills and speech and language performance in young children with cleft lip and palate. The Cleft Palate–Craniofacial Journal      48,     301–11.

Chapman, K. L., & Willadsen, E. (2011). The development of speech in children with cleft palate. Cleft palate speech: Assessment and intervention, 23-40.

D’Antonio, L.L. and Scherer, N.J., 1995. The evaluation of speech disorders associated with clefting. Cleft palate speech management: A multidisciplinary approach, pp.176-220.

Dobbelsteyn, C., Bird, E. K. R., Parker, J., Griffiths, C., Budden, A., Flood, K., Stilson, A., 2014, Effectiveness of the corrective babbling speech treatment program for children with a history of cleft palate or velopharyngeal dysfunction. Cleft Palate-Craniofacial Journal, 51(2), 129-144.

Ha, S. (2015). Effectiveness of a parent-implemented intervention program for young children with cleft palate. International Journal Of Pediatric Otorhinolaryngology, 79(5), 707-715.

Hardin-Jones, M. and Chapman, K.L., 2014. Early lexical characteristics of toddlers with cleft lip and palate. The Cleft palate-craniofacial journal, 51(6), pp.622-631.

Harding, A. & Bryan, A. (2000). The use of multi-sensory input modelling to stimulate speech output processing: A teaching and demonstration video [Video]. Available from Cleft.NET.East, Box 46, Addenbrookes Hospital, Cambridge University Hospitals Foundation Trust, Cambridge, UK.

Harding-Bell, A., & Howard, S. (2011). Phonological approaches to speech difficulties associated with cleft palate. Cleft Lip and Palate: speech assessment, analysis and intervention, Chichester: John Wiley and Sons.

Kummer, A.W. (2011) Speech therapy for errors secondary to cleft palate and velopharyngeal dysfunction. Seminars in speech and language, 32(2): 191-198.

Lancaster, H.S., Lien, K.M., Chow, J.C., Frey, J.R., Scherer, N.J. and Kaiser, A.P., 2020. Early speech and language development in children with nonsyndromic cleft lip and/or palate: A meta-analysis. Journal of Speech, Language, and Hearing Research, 63(1), pp.14-31.

McLeod, S., Harrison, L.J. and McCormack, J., 2012. The intelligibility in context scale: Validity and reliability of a subjective rating measure. Journal of Speech, Language, and Hearing Research.

Morgan, A.R., Bellucci, C.C., Coppersmith, J., Linde, S.B., Curtis, A., Albert, M., O’Gara, M.M. and Kapp-Simon, K., 2017. Language Development in Children With Cleft Palate With or Without Cleft Lip Adopted From Non–English-Speaking Countries. American Journal of Speech-Language Pathology, 26(2), pp.342-354.

Pamplona, M. C., Ysunza, A., & Espinosa, J. (1999). A comparative trial of two modalities of speech intervention for compensatory articulation in cleft palate children, phonologic approach versus articulatory approach. International journal of pediatric otorhinolaryngology, 49(1), 21-26.

Peterson-Falzone, S.J., Trost- Cardamone, J.E., Karnell, M.P., & Hardin-Jones, M.A. (2006). The Clinician’s Guide to Treating Cleft Palate Speech. US: Mosby Elsevier.

Richman, L.C., 1980. Cognitive patterns and learning disabilities in cleft palate children with verbal deficits. Journal of Speech, Language, and Hearing Research, 23(2), pp.447-456.

Scherer, N. J., D’antonio, L. L.,and McGahey, H. (2008b). Early intervention for speech impairment in children with cleft palate. Cleft Palate- Craniofacial Journal, 45(1), 18-31.

Scherer, N. J. (1999). The speech and language status of toddlers with cleft lip and/or palate following early vocabulary intervention. American Journal of Speech-Language Pathology, 8(1), 81-93.

Sell, D. and Pereira, V., 2011. Instrumental/speech imaging to analyse structure and function. Cleft Lip and Palate: speech assessment, analysis and intervention, Chichester: John Wiley and Sons.

Sell, D., Harding, A., & Grunwell, P. (1999). GOS. SP. ASS.’98: an assessment for speech disorders associated with cleft palate and/or velopharyngeal dysfunction (revised). International Journal of

Sell, D., Mars, M., & Worrell, E. (2006) Process and outcome study of multidisciplinary prosthetic treatment for velopharyngeal dysfunction. International Journal of Language and Communication Disorders, 41(5): 495-511.

Sweeney, T.  (2011). Nasality assessment and intervention. In S. J. Howard & A. Lohmander (Eds.) Cleft palate speech: Assessment and intervention (pp.199-220). Oxford: John Wiley & Sons Ltd.

Sweeney T, Hegarty F, Powell K, Deasy L, O’Regan M, Sell D. (in press). A Randomised Controlled Trial Comparing Parent Led Therapist Supervised Articulation Therapy (PLAT) with Routine Intervention for Children with speech disorders associated with cleft palate. International Journal for Language and Communication Disorders.

Watkins, S.E., Meyer, R.E., Aylsworth, A.S., Marcus, J.R., Allori, A.C., Pimenta, L., Lipinski, R.J. and Strauss, R.P., 2018. Academic achievement among children with nonsyndromic orofacial clefts: A population-based study. The Cleft Palate-Craniofacial Journal, 55(1), pp.12-20.

Willadsen, E. (2007). From babbling to meaningful speech in Danish children born with and without cleft lip and palate. Unpublished doctoral dissertation]. University of Aarhus, Aarhus, Denmark.

Williams, A. L. (2000). Multiple oppositions: case studies of variables in phonological intervention. American Journal of Speech-Language Pathology, 9(4), 289-299.

Young, S. E., Purcell, A. A., & Ballard, K. J. (2010). Expressive language skills in Chinese Singaporean preschoolers with nonsyndromic cleft lip and/or palate. International Journal of Pediatric Otorhinolaryngology, 74(5), 456-464.

How can babies with unrepaired Cleft Lip and/or Palate be supported during feeding?

Cleft Lip and Palate Association (2015). Feeding. Retrieved from https://www.clapa.com/treatment/feeding/

Miller, C.K. (2011). Feeding issues and interventions in infants and children with clefts and craniofacial syndromes. Seminars in Speech and Language, 32(2): 115-126.

Cleft Palate Foundation (2009) Feeding your baby. Available online: cleftlin.org/docs/Booklets/FDG-01.pdf