Semin Speech Lang 2011; 32(2): 127-140
DOI: 10.1055/s-0031-1277715
© Thieme Medical Publishers

Cognitive and Language Issues Associated with Cleft Lip and Palate

Mary Hardin-Jones1 , Kathy L. Chapman2
  • 1University of Wyoming, Laramie, Wyoming
  • 2University of Utah, Salt Lake City, Utah
Further Information

Publication History

Publication Date:
26 September 2011 (online)

ABSTRACT

Language and cognitive performance of children with cleft palate with/without cleft lip (CP ± L) have received relatively little attention in the literature to date. The lack of attention directed toward these areas is probably attributed to two factors. First, the speech characteristics associated with velopharyngeal inadequacy are undoubtedly the most salient problems encountered by clinicians treating these children and thus have taken priority in research for many years. Second, early findings suggesting little difference between these children and their typically developing peers in language and cognition simply discouraged additional study. More recent investigations, however, have expanded the scope of study to include toddlers and adults and utilized alternative methodologies to study this population. In this article, we will summarize the literature and explore recent findings associated with language and cognition in children with CP ± L. In addition, we will also examine performance of children with velocardiofacial syndrome—a common syndrome associated with CP ± L.

REFERENCES

  • 1 Peterson-Falzone S J, Hardin-Jones M A, Karnell M P. Cleft Palate Speech. 4th edition. St. Louis, MO: Elsevier; 2010
  • 2 Fox D, Lynch J, Brookshire B. Selected developmental factors of cleft palate children between two and thirty-three months of age.  Cleft Palate J. 1978;  15 (3) 239-245
  • 3 Neiman G S, Savage H E. Development of infants and toddlers with clefts from birth to three years of age.  Cleft Palate Craniofac J. 1997;  34 (3) 218-225
  • 4 Bayley N. Bayley Scales of Infant Development. New York, NY: Psychological Corp; 1969
  • 5 Broen P A, Devers M C, Doyle S S, Prouty J M, Moller K T. Acquisition of linguistic and cognitive skills by children with cleft palate.  J Speech Lang Hear Res. 1998;  41 (3) 676-687
  • 6 Jocelyn L J, Penko M A, Rode H L. Cognition, communication, and hearing in young children with cleft lip and palate and in control children: a longitudinal study.  Pediatrics. 1996;  97 (4) 529-534
  • 7 Kapp-Simon K A, Krueckeberg S. Mental development in infants with cleft lip and/or palate.  Cleft Palate Craniofac J. 2000;  37 (1) 65-70
  • 8 Speltz M L, Endriga M C, Hill S, Maris C L, Jones K, Omnell M L. Cognitive and psychomotor development of infants with orofacial clefts.  J Pediatr Psychol. 2000;  25 (3) 185-190
  • 9 Starr P, Chinsky R, Canter H, Meier J. Mental, motor, and social behavior of infants with cleft lip and/or cleft palate.  Cleft Palate J. 1977;  14 (2) 140-147
  • 10 Endriga M C, Kapp-Simon K A. Psychological issues in craniofacial care: state of the art.  Cleft Palate Craniofac J. 1999;  36 (1) 3-11
  • 11 Richman L C, Nopoulos P. Neuropsychological and neuroimaging aspects of cleft lip and palate. In: Losee J, Kirschner R, eds. Comprehensive Cleft Care. New York, NY: McGraw-Hill; 2008
  • 12 Broder H L, Richman L C, Matheson P B. Learning disability, school achievement, and grade retention among children with cleft: a two-center study.  Cleft Palate Craniofac J. 1998;  35 (2) 127-131
  • 13 Richman L C, Eliason M J, Lindgren S D. Reading disability in children with clefts.  Cleft Palate J. 1988;  25 (1) 21-25
  • 14 Richman L C, Ryan S M. Do the reading disabilities of children with cleft fit into current models of developmental dyslexia?.  Cleft Palate Craniofac J. 2003;  40 (2) 154-157
  • 15 Richman L C, Wilgenbusch T, Hall T. Spontaneous verbal labeling: visual memory and reading ability in children with cleft.  Cleft Palate Craniofac J. 2005;  42 (5) 565-569
  • 16 Vellutino F R, Fletcher J M, Snowling M J, Scanlon D M. Specific reading disability (dyslexia): what have we learned in the past four decades?.  J Child Psychol Psychiatry. 2004;  45 (1) 2-40
  • 17 Collett B R, Leroux B, Speltz M L. Language and early reading among children with orofacial clefts.  Cleft Palate Craniofac J. 2010;  47 (3) 284-292
  • 18 Wilson K. Phonological awareness in children with clefts.  Presented at: the Annual Meeting of the American Speech-Language-Hearing Association; November 15–17, 2007; Boston, MA
  • 19 Collett B R, Stott-Miller M, Kapp-Simon K A, Cunningham M L, Speltz M L. Reading in children with orofacial clefts versus controls.  J Pediatr Psychol. 2010;  35 (2) 199-208
  • 20 Chapman K. The relationship between early reading skills and speech and language performance in young children with cleft lip and palate.  Cleft Palate Craniofac J. 2011;  In press
  • 21 Reid D K, Hresko W P, Hammill D D. Test of Early Reading Ability. 3rd ed. Austin, TX: PRO-ED; 2001
  • 22 Goldman R, Fristoe M. The Goldman-Fristoe Test of Articulation. 2d ed. Circle Pines, MN: American Guidance Service; 2000
  • 23 Newcomer P L, Hammill D D. Test of Language Development-Primary. 3rd ed. Austin, TX: PRO-ED; 1997
  • 24 Nopoulos P, Berg S, VanDemark D, Richman L, Canady J, Andreasen N C. Cognitive dysfunction in adult males with non-syndromic clefts of the lip and/or palate.  Neuropsychologia. 2002;  40 (12) 2178-2184
  • 25 Wechsler D. Wechsler Adult Intelligence Scale-Revised. New York, NY: The Psychological Corporation; 1981
  • 26 Estes R E, Morris H L. Relationships among intelligence, speech proficiency, and hearing sensitivity in children with cleft palates.  Cleft Palate J. 1970;  7 763-773
  • 27 Goodstein L D. Intellectual impairment in children with cleft palates.  J Speech Hear Res. 1961;  4 287-294
  • 28 Lamb M M, Wilson F B, Leeper H A. The intellectual function of cleft palate children compared on the basis of cleft type and sex.  Cleft Palate J. 1973;  10 367-377
  • 29 Richman L C. Cognitive patterns and learning disabilities of cleft palate children with verbal deficits.  J Speech Hear Res. 1980;  23 (2) 447-456
  • 30 Richman L C, Eliason M J. Type of reading disability related to cleft type and neuropsychological patterns.  Cleft Palate J. 1984;  21 (1) 1-6
  • 31 Leeper Jr H A, Pannbacker M, Roginski J. Oral language characteristics of adult cleft-palate speakers compared on the basis of cleft type and sex.  J Commun Disord. 1980;  13 (2) 133-146
  • 32 Morris H L. Communication skills of children with cleft lips and palates.  J Speech Hear Res. 1962;  5 79-90
  • 33 Nation J E. Vocabulary comprehension and usage of preschool cleft palate and normal children.  Cleft Palate J. 1970;  7 639-644
  • 34 Philips B J, Harrison R J. Language skills of preschool cleft palate children.  Cleft Palate J. 1969;  6 108-119
  • 35 Spriestersbach D C, Darley F L, Morris H L. Language skills in children with cleft palates.  J Speech Hear Res. 1958;  1 (3) 279-285
  • 36 Long N V, Dalston R M. Comprehension abilities of one-year-old infants with cleft lip and palate.  Cleft Palate J. 1983;  20 (4) 303-306
  • 37 Scherer N J, D'Antonio L. Parent questionnaire for screening early language development in children with cleft palate.  Cleft Palate Craniofac J. 1995;  32 7-13
  • 38 Snyder L E, Scherer N J. The development of symbolic play and language in toddlers with cleft palate.  Am J Speech Lang Pathol. 2004;  13 (1) 66-80
  • 39 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 (4) 400-404
  • 40 Long N V, Dalston R M. Gestural communication in twelve-month-old cleft lip and palate children.  Cleft Palate J. 1982;  19 (1) 57-61
  • 41 Long N V, Dalston R M. Paired gestural and vocal behavior in one-year-old cleft lip and palate children.  J Speech Hear Disord. 1982;  47 (4) 403-406
  • 42 Chapman K L, Hardin-Jones M A, Goldstein J A, Halter K A, Havlik R J, Schulte J. Timing of palatal surgery and speech outcome.  Cleft Palate Craniofac J. 2008;  45 (3) 297-308
  • 43 Parent Report Measures .FIRST WORDS Project. The Florida State University Research Foundation 2007. Available at: http://firstwords.fsu.edu/parentsReportMeasures.html# Accessed October 10, 2010
  • 44 Chapman K L, Hardin-Jones M A. Consideration of Individual Differences in Speech Development, Outcome, and Management: Children with Cleft Lip and Palate. In: Weiss A, ed. Perspectives on Individual Differences Affecting Therapeutic Change in Communication Disorders. Florence, KY: Taylor & Francis/Psychology Press; 2010
  • 45 Scarborough H. Index of productive syntax.  Appl Psycholinguist. 1990;  11 1-22
  • 46 Scherer N J, D'Antonio L. Language and play development in toddlers with cleft lip and/or palate.  Am J Speech Lang Pathol. 1997;  6 48-54
  • 47 Morris H, Ozanne A. Phonetic, phonological, and language skills of children with a cleft palate.  Cleft Palate Craniofac J. 2003;  40 (5) 460-470
  • 48 Chapman K L, Hardin M A. Communication competence in children with cleft lip and palate. In: Bardach J, Morris H L, eds. Multidisciplinary Management of Cleft Lip and Palate. Philadelphia, PA: W.B. Saunders; 1990
  • 49 Fey M E. Language Intervention with Young Children. San Diego, CA: College-Hill Press; 1986
  • 50 Chapman K L, Graham K T, Gooch J, Visconti C. Conversational skills of preschool and school-age children with cleft lip and palate.  Cleft Palate Craniofac J. 1998;  35 (6) 503-516
  • 51 Warr-Leeper G, Crone L, Carruthers A, Leeper H. A comparison of the performance of preschool children with cleft lip and/or palate on the Test of Pragmatic Skills.  Presented at: the Annual Meeting of the American Cleft Palate Association; April 1988; Williamsburg, VA
  • 52 Frederickson M S, Chapman K L, Hardin-Jones M. Conversational skills of children with cleft lip and palate: a replication and extension.  Cleft Palate Craniofac J. 2006;  43 (2) 179-188
  • 53 Faircloth S, Faircloth M. Delayed language and linguistics variations. In: Grabb W, Rosenstein S, Bzoch K, eds. Cleft Lip and Palate. Boston, MA: Little Brown; 1971
  • 54 Morris H L. Etiological bases for speech problems. In: Spriestersbach D C, Sherman D, eds. Cleft Palate and Communication. New York, NY: Academic Press; 1968: 119-168
  • 55 Vallino L D, Zuker R, Napoli J A. A study of speech, language, hearing, and dentition in children with cleft lip only.  Cleft Palate Craniofac J. 2008;  45 (5) 485-494
  • 56 Zimmerman I L, Steiner V G, Pond R E. Preschool Language Scale-Revised. San Antonio, TX: The Psychological Corporation; 1979
  • 57 Tomblin J B, Records N L, Buckwalter P, Zhang X, Smith E, O'Brien M. Prevalence of specific language impairment in kindergarten children.  J Speech Lang Hear Res. 1997;  40 (6) 1245-1260
  • 58 Newcomer P L, Hammill D D. Test of Language Development-Primary. Austin, TX: PRO-ED; 1988
  • 59 Hammill D D, Newcomer P L. Test of Language Development-Intermediate. Austin, TX: PRO-ED; 1988
  • 60 Nash P, Stengelhofen J, Toombs L, Brown J, Kellow B. National survey of children aged with persisting communication problems associated with cleft palate.  Child Lang Teach Ther. 2001;  17 19-34
  • 61 Peterson-Falzone S J. Speech outcomes in adolescents with cleft lip and palate.  Cleft Palate Craniofac J. 1995;  32 (2) 125-128
  • 62 Richman L, Nopoulos P. Neuropsychological and neuroimaging aspects of cleft lip and palate. In: Losee J E, Kirschner R E, eds. Comprehensive Cleft Care. New York, NY: McGraw- Hill; 2009
  • 63 Conrad A L, 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
  • 64 Pannbacker M. Oral language skills of adult cleft palate speakers.  Cleft Palate J. 1975;  12 (00) 95-106
  • 65 Estes R E, Morris H L. Relationships among intelligence, speech proficiency, and hearing sensitivity in children with cleft palates.  Cleft Palate J. 1970;  7 763-773
  • 66 Ceponiene R, Hukki J, Cheour M, Haapanen M L, Ranta R, Näätänen R. Cortical auditory dysfunction in children with oral clefts: relation with cleft type.  Clin Neurophysiol. 1999;  110 (11) 1921-1926
  • 67 Ceponiene R, Hukki J, Cheour M et al.. Dysfunction of the auditory cortex persists in infants with certain cleft types.  Dev Med Child Neurol. 2000;  42 (4) 258-265
  • 68 Cheour M, Haapanen M L, Ceponiene R, Hukki J, Ranta R, Näätänen R. Mismatch negativity (MMN) as an index of auditory sensory memory deficit in cleft-palate and CATCH syndrome children.  Neuroreport. 1998;  9 (12) 2709-2712
  • 69 Cheour M, Ceponiene R, Hukki J, Haapanen M L, Näätänen R, Alho K. Brain dysfunction in neonates with cleft palate revealed by the mismatch negativity.  Clin Neurophysiol. 1999;  110 (2) 324-328
  • 70 Nopoulos P, Berg S, VanDemark D, Richman L, Canady J, Andreasen N C. Increased incidence of a midline brain anomaly in patients with nonsyndromic clefts of the lip and/or palate.  J Neuroimaging. 2001;  11 (4) 418-424
  • 71 Nopoulos P, Berg S, Canady J, Richman L, Van Demark D, Andreasen N C. Structural brain abnormalities in adult males with clefts of the lip and/or palate.  Genet Med. 2002;  4 (1) 1-9
  • 72 Boes A D, Murko V, Wood J L et al.. Social function in boys with cleft lip and palate: relationship to ventral frontal cortex morphology.  Behav Brain Res. 2007;  181 (2) 224-231
  • 73 Nopoulos P, Choe I, Berg S, Van Demark D, Canady J, Richman L. Ventral frontal cortex morphology in adult males with isolated orofacial clefts: relationship to abnormalities in social function.  Cleft Palate Craniofac J. 2005;  42 (2) 138-144
  • 74 Shriver A S, Canady J, Richman L, Andreasen N C, Nopoulos P. Structure and function of the superior temporal plane in adult males with cleft lip and palate: pathologic enlargement with no relationship to childhood hearing deficits.  J Child Psychol Psychiatry. 2006;  47 (10) 994-1002
  • 75 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 (6) 683-690
  • 76 Nopoulos P, Langbehn D R, Canady J, Magnotta V, Richman L. Abnormal brain structure in children with isolated clefts of the lip or palate.  Arch Pediatr Adolesc Med. 2007;  161 (8) 753-758
  • 77 van der Plas E, Conrad A, Canady J, Richman L, Nopoulos P. Effects of unilateral clefts on brain structure.  Arch Pediatr Adolesc Med. 2010;  164 (8) 763-768
  • 78 Scambler P J. The 22q11 deletion syndromes.  Hum Mol Genet. 2000;  9 (16) 2421-2426
  • 79 Goldberg R, Motzkin B, Marion R, Scambler P J, Shprintzen R J. Velo-cardio-facial syndrome: a review of 120 patients.  Am J Med Genet. 1993;  45 (3) 313-319
  • 80 Murphy K C, Jones L A, Owen M J. High rates of schizophrenia in adults with velo-cardio-facial syndrome.  Arch Gen Psychiatry. 1999;  56 (10) 940-945
  • 81 Golding-Kushner K J, Weller G, Shprintzen R J. Velo-cardio-facial syndrome: language and psychological profiles.  J Craniofac Genet Dev Biol. 1985;  5 (3) 259-266
  • 82 Moss E M, Batshaw M L, Solot C B et al.. Psychoeducational profile of the 22q11.2 microdeletion: a complex pattern.  J Pediatr. 1999;  134 (2) 193-198
  • 83 Swillen A, Devriendt K, Legius E et al.. Intelligence and psychosocial adjustment in velocardiofacial syndrome: a study of 37 children and adolescents with VCFS.  J Med Genet. 1997;  34 (6) 453-458
  • 84 Zinkstok J, van Amelsvoort T. Neuropsychological profile and neuroimaging in patients with 22Q11.2 deletion syndrome: a review.  Child Neuropsychol. 2005;  11 (1) 21-37
  • 85 Shprintzen R J. Velo-cardio-facial syndrome: a distinctive behavioral phenotype.  Ment Retard Dev Disabil Res Rev. 2000;  6 (2) 142-147
  • 86 Antshel K M, Kates W R, Roizen N, Fremont W, Shprintzen R J. 22q11.2 deletion syndrome: genetics, neuroanatomy and cognitive/behavioral features keywords.  Child Neuropsychol. 2005;  11 (1) 5-19
  • 87 De Smedt B, Swillen A, Devriendt K, Fryns J P, Verschaffel L, Ghesquiere P. Mathematical disabilities in young primary school children with velo-cardio-facial syndrome.  Genet Couns. 2006;  17 (3) 259-280
  • 88 De Smedt B, Swillen A, Devriendt K et al.. Cognitive correlates of mathematical disabilities in children with velo-cardio-facial syndrome.  Genet Couns. 2008;  19 (1) 71-94
  • 89 Lajiness-O'Neill R, Beaulieu I, Asamoah A Lajiness-O-Neill R et al. The neuropsychological phenotype of velocardiofacial syndrome (VCFS): relationship to psychopathology.  Arch Clin Neuropsychol. 2006;  21 (2) 175-184
  • 90 Scherer N J, D'Antonio L L, Kalbfleisch J H. Early speech and language development in children with velocardiofacial syndrome.  Am J Med Genet. 1999;  88 (6) 714-723
  • 91 D'Antonio L L, Scherer N J, Miller L L, Kalbfleisch J H, Bartley J A. Analysis of speech characteristics in children with velocardiofacial syndrome (VCFS) and children with phenotypic overlap without VCFS.  Cleft Palate Craniofac J. 2001;  38 (5) 455-467
  • 92 Solot C B, Gerdes M, Kirschner R E et al.. Communication issues in 22q11.2 deletion syndrome: children at risk.  Genet Med. 2001;  3 (1) 67-71
  • 93 Gerdes M, Solot C, Wang P P et al.. Cognitive and behavior profile of preschool children with chromosome 22q11.2 deletion.  Am J Med Genet. 1999;  85 (2) 127-133

1 Peterson-Falzone et al[1] pointed out that early investigations did not always sort findings by cleft type nor exclude syndromes and so only more recent investigations will be reported here.

2 The study by Chapman[20] administered the Test of Early Reading-3,[21] which assesses early reading skills.

3 22q11.2 deletion is believed to be responsible for velocardiofacial syndrome as well as several other previously described syndromes. Therefore, VCFS is also called 22q11.2 deletion syndrome.

4 Children with VCFS are delayed in the acquisition of other developmental milestones as well, including sitting alone, crawling, and walking.[1]

Mary Hardin-JonesPh.D. 

Division of Communication Disorders, Dept. 3311, University of Wyoming

1000 E. University Avenue, Laramie, WY 82072

Email: mhardinj@uwyo.edu

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