A prospective cross-sectional study of speech in patients with the 22q11 deletion syndrome

Persson, Christina; Lohmander, Anette; Jönsson, Robert; Óskarsdóttir, Sólveig; Söderpalm, Ewa

doi:10.1016/s0021-9924(02)00133-8

Cited by 47 publications

(68 citation statements)

References 39 publications

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“…The most common types of clefts associated with the deletion are overt, submucous or occult submucous cleft of the secondary palate [10]. The most common type of cleft in this study was submucous cleft palate, found in almost 20% of the children.…”

Section: Discussionmentioning

confidence: 51%

Presenting phenotype in 100 children with the 22q11 deletion syndrome

Óskarsdóttir¹,

et al. 2004

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Section: Discussionmentioning

confidence: 51%

Presenting phenotype in 100 children with the 22q11 deletion syndrome

Óskarsdóttir¹,

et al. 2004

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Section: Articulation Skills In Children With Vcfsmentioning

confidence: 77%

“…Persson et al (2003) and Golding-Kushner (2001, 2005 have reported systematic speech errors similar to the developmental phonological processes often observed in young children with typical speech development. In addition, studies also report a high percentage of glottal stops in the early speech productions of young children with VCFS Scherer et al, 1999;Golding-Kushner, 1991, 2005Persson et al, 2003). Researchers have not yet found a direct relationship between velopharyngeal factors (e.g., extent of velopharyngeal inadequacy, age at time of palatal repair, type or timing of secondary speech surgery) and measures of articulation skills in children with VCFS , Scherer et al, 1999.…”

Section: Articulation Skills In Children With Vcfsmentioning

confidence: 77%

“…Persson et al (2003) and Golding-Kushner (2001, 2005 Scherer et al, 1999; Golding-Kushner, 1991, 2005Persson et al, 2003 et al, , Scherer et al, 1999. Differences in the ages of participants across studies, as well as variation in speech tasks utilized (e.g.…”

Section: Articulation Skills In Children With Vcfsmentioning

confidence: 99%

“…Children with VCFS often display persistent hypernasality and articulation skills that are significantly below those of their age peers. In addition, children with VCFS may demonstrate systematic phonological errors, dysarthria, developmental apraxia of speech, voice disorders, or a combination of these (Kummer et al, 2007;Scherer et al, 1999;Carneol et al, 1999;Persson et al, 2003;Golding-Kushner 1995, 2005. Although children with VCFS may demonstrate speech that is in some ways similar to that of children with non-syndromic cleft palate or VPD, children with VCFS, regardless of the presence of clefting, have consistently been shown to have more impaired articulation skills than these children (Scherer et al, 1999;Scherer et al, 2001;D'Antonio et al, 2001; Golding-Kushner, 1991;DeMarco, 2004DeMarco, , 2005.…”

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confidence: 99%

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Factors Affecting Articulation Skills in Children with Velocardiofacial Syndrome and Children with Cleft Palate or Velopharyngeal Dysfunction: A Preliminary Report

Baylis

Munson

Moller

2008

The Cleft Palate Craniofacial Journal

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Objective-To examine the influence of speech perception, cognition, and implicit phonological learning on articulation skills of children with Velocardiofacial syndrome (VCFS) and children with cleft palate or velopharyngeal dysfunction (VPD). Design-Cross-sectional group experimental design.Participants-8 children with VCFS and 5 children with non-syndromic cleft palate or VPD.Methods and Measures-All children participated in a phonetic inventory task, speech perception task, implicit priming nonword repetition task, conversational sample, nonverbal intelligence test, and hearing screening. Speech tasks were scored for percentage of phonemes correctly produced. Group differences and relations among measures were examined using nonparametric statistics.Results-Children in the VCFS group demonstrated significantly poorer articulation skills and lower standard scores of nonverbal intelligence compared to the children with cleft palate or VPD. There were no significant group differences in speech perception skills. For the implicit priming task, both groups of children were more accurate in producing primed nonwords than unprimed nonwords. Nonverbal intelligence and severity of velopharyngeal inadequacy for speech were correlated with articulation skills.Conclusions-In this study, children with VCFS had poorer articulation skills compared to children with cleft palate or VPD. Articulation difficulties seen in the children with VCFS did not appear to be associated with speech perception skills or the ability to learn new phonological representations. Future research should continue to examine relationships between articulation, Address Correspondence to: Adriane Baylis, Ph.D., Masters Family Speech and Hearing Clinic, Children's Hospital of Wisconsin, P.O. Box 1997, Milwaukee, WI, 53201-1997 NIH Public Access Author ManuscriptCleft Palate Craniofac J. Author manuscript; available in PMC 2010 August 16. NIH-PA Author ManuscriptNIH-PA Author Manuscript NIH-PA Author Manuscript cognition, and velopharyngeal dysfunction in a larger sample of children with cleft palate and VCFS. KeywordsVelocardiofacial syndrome; 22q11.2 deletion; articulation; cleft palate; velopharyngeal dysfunction Velocardiofacial syndrome (VCFS) is a relatively common multianomaly syndrome characterized by cleft palate, velopharyngeal dysfunction, speech-language disorders, cardiac anomalies, cognitive-behavioral disorders, characteristic facial features, differences in brain morphology, and a variety of other health and psychosocial problems (Shprintzen et al., 1978: Shprintzen et al., 1981Goldberg et al., 1993;Golding-Kushner et al., 1985;Golding-Kushner, 2005; McDonald-McGinn et al., 1997;Eliez et al., 2001). VCFS is a genetic disorder associated with a microdeletion at chromosome 22q11.2, assessed by fluorescent in situ hybridization (FISH) testing (Driscoll et al., 1992;Scambler et al., 1992). Children with VCFS are often initially encountered and diagnosed through a Cleft Palate/ Craniofacial Team, likely due to the high incidence o...

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