t in cri du chat syndrome - universitetet i oslo...background: cri du chat syndrome (cdcs) •...

TEMPLATES IN CRI DU CHAT SYNDROME

Kristian E. Kristoffersen1, Nina Gram Garmann2 and Hanne Gram Simonsen1 1Department of Linguistics and Scandinavian Studies, University of Oslo 2Oslo and Akershus University College of Applied Sciences NorPhLex Workshop, Stockholm, 15 November 2012

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Overview

•  Background –  Cri du chat syndrome –  Templates –  Other measures (PPC, PWP, variegation)

•  Method •  Analysis •  Discussion and conclusion

Background: Cri du chat syndrome (CdCS) •  Genetic disorder

–  Cause: partial deletion of material on the short arm of chromosome 5

–  Incidence: between 1:15,000 and 1:50,000 births

•  Some clinical features –  High-pitched cry in infancy and childhood –  Short attention span –  Impaired fine and gross motor skills –  Mild to profound intellectual disability –  Delayed speech and language development

Background: language skills in CdCS

•  Receptive language skills are better than productive language skills •  Syntax: short MLU, omissions, deviant word order and prefabricated

units •  Morphology: relatively well preserved inflectional morphology; some

overgeneralizations indicate internalized schemas •  Speech sounds:

–  Small consonant inventories and frequent misarticulations –  Patterns of deviation:

•  Few coronal consonants •  Prenasalised consonants •  No systematic distinction between voiced and voiceless consonants

Background: Templates

Ny Powerpoint mal 2011

Vihman & Velleman (2000): “Different children arrive at different solutions to (1) the conflict between their phonetic skills and the demands of the ambient language and (2) the representational problem posed by the learning of a large and arbitrary set of patterned sound-meaning pairs (words and/or phrases)”

Background: Templates

•  Templates are generalizations over known words that are –  closely related to the child’s babbling patterns –  constrained by the child’s articulatory skills

•  Templates are related to two types of words: –  Selected words: target words that are close to the

template –  Adapted words: target words that are NOT close

to the target, but are adapted to it

7

Templates: Example

•  Harmony: ˈC1VC1V – Selected:

• pappa ’daddy’ [¹pæpæ] •  kake ‘cake’ [ˈkakʰæʰ]

– Adapted: •  lampa ’the lamp’ [ˈpəәpʰa ̀]

Templates: Example from a boy with cri du chat syndrome Target P3 (boy aged 9;2) mamma ‘mommy’ mʌmʌ nisse ‘gnome’ ɡiɡi Pippi ‘name’ pipi pippip ‘pippip’ pəәpəә Lisbeth ‘name’ ŋɡaŋɡa Oddbjørn ‘name’ ʔɔ̃ʔɔ̃ Ole Brum ‘Winnie the Pooh’ ʔɔ̃ʔɔ̃

•  TEMPLATE: Harmony – C1VC1V •  Selected form: mʌmʌ ‘mommy’, pipi ‘Pippi [name]’, pəәpəә ‘pippip [bird]’ •  Adapted forms: gigi ‘gnome’, ŋɡaŋɡa ‘Lisbeth [name]’, ʔɔ̃ʔɔ̃ ‘Oddbjørn [name]’, ʔɔ̃ʔɔ̃ ‘Winnie the Pooh’

Templates: Longitudinal example from a girl with cri du chat syndrome

Target 4;6 5;9

pappa ‘daddy’ ˈpæpəә ˈpæpæ

bade ‘take a bath’ ˈpæpəә ˈpatəә

gaffel ‘fork’ ˈcæcəә ˈæti

eple ‘apple’ ˈpæpæ ˈæpæ

•  TEMPLATE: Harmony – C1VC1V (at 4;6) •  Selected form: ˈpæpəә ‘daddy’ •  Adapted forms: ˈpæpəә ‘take a bath’, ˈcæcəә ’gaffel’,

ˈpæpæ ‘apple’

Questions

1.  Do older childen with cri du chat syndrome use templates that are normally associated with far younger typically developing children?

2.  Does the template score correlate with other measures?

3.  If there are such correlations, how can we interpret them, and what do they tell us about templates?

Method: Participants

ID Gender Age Utterances Intelligibility P1 M 10;0 One-word poor P2 F 10;8 Multi-word poor P3 M 9;2 One-word poor P4 F 12;3 One-word poor P5 F 5;4 Multi-word medium P6 F 6;4 Multi-word good P7 F 6;9 Multi-word medium P8 F 4;6 One-word poor

Example pronunciations

ID Pronunciation Target Gloss Intelligibility

P1 ˈʔa.ˈʔa.ˈpa ˈbaskətˌbaɭ ‘basket ball’ poor

P2 fəlacˡ fɭag ‘flag’ poor

P3 n ͡gu sku: ‘shoe’ poor

P4 pæˈpa eple ‘apple’ poor

P5 kˡol sko:l ‘saucer’ medium

P6 ˈtı:ge ˈsti:ge ‘ladder’ good

P7 ˈbləlɛ ˈbɾilɾ̩ ‘glasses’ medium

P8 ˈpæpæ ˈeple ‘apple’ poor

Method: Procedure and analysis

•  Picture and object naming •  Transcription:

– Narrow IPA – Transcription agreement: 71%.

Method: Measures

•  Template score •  Consonant inventory •  Percent consonants correct (PCC-R) •  Variegation •  Proximity to target pronunciation (PWP)

RESULTS

Template scores

•  Scale from 0–100 % •  Minimum score:

all productions are target like •  Maximum score:

all productions are adapted to specific and stable templates

Template scores

ID Age Template Score (%)

CI size non-deviant

P4 12;3 55 15

P5 5;4 51 17

P3 9;2 50 9

P8 4;6 33 9

P1 10;0 28 6

P2 10;8 17 13

P7 6;9 9 18

P6 6;4 6 18

Template scores

ID Age Template Score CI size non-deviant

P4 12;3 55 15

P5 5;4 51 17

P3 9;2 50 9

P8 4;6 33 9

P1 10;0 28 6

P2 10;8 17 13

P7 6;9 9 18

P6 6;4 6 18

Template scores

ID Age Template Score (%)

CI size non-deviant

P4 12;3 55 15

P5 5;4 51 17

P3 9;2 50 9

P8 4;6 33 9

P1 10;0 28 6

P2 10;8 17 13

P7 6;9 9 18

P6 6;4 6 18

PCC-R

•  Percent Consonants Correct Revised (Shriberg et al 1997)

•  Proportion of correctly produced consonants: –  Deletions and substitutions are scored as

incorrect –  Distorted consonants are scored as correct


PCC-R

ID Age Template score (%)

PCC-R

P6 6;4 6 93

P7 6;9 9 83

P2 10;8 17 72

P5 5;4 51 52

P4 12;3 55 49

P3 9;2 50 35

P8 4;6 33 23

P1 10;0 28 2

Variegation

•  Scale from 1–3 •  1: the production consists of only vowels,

glides, or /h/, e.g. [a], [ja] or [ha] •  2: the production consists of only one

consonant, e.g. [ma] or [mama] •  3: the production consists of more than one

consonant, e.g. [kat], [site]


Variegation

ID Age Template score Variegation score

P2 10;8 17 3.00

P6 6;4 6 2.76

P7 6;9 9 2.62

P4 12;3 55 2.35

P5 5;4 51 2.16

P3 9;2 50 1.84

P8 4;6 33 1.64

P1 10;0 28 1.14

Proportion of Whole-word Proximity (PWP) •  Measures degree of similarity to target words •  Based on Ingram’s (2002) Phonological

Mean Length of Utterance: –  NUMBER OF CONSONANTS + NUMBER OF VOWELS +

NUMBER OF CORRECT CONSONANTS –  ba for ball have a PMLU of 1+1+1=3 –  Target PMLU: 2+1+2=5

Proportion of Whole-word Proximity (PWP) •  Measures degree of similarity to target

words •  PWP = CHILD PMLU / TARGET PMLU

– PWP for ba: 3 / 5 = 60%

Proximity to target words (PWP)

ID Age Template score PWP (%)

P6 6;4 6 93

P7 6;9 9 89

P2 10;8 17 74

P4 12;3 55 74

P5 5;4 51 66

P3 9;2 50 47

P8 4;6 33 45

P1 10;0 28 38

Why are P1 and P8 different?

•  Templates are –  Early phonological representations –  Expressions of articulatory skills

•  P1 and P8 have poor articulatory skills •  They responded to all the items in the

elicitation task => rich lexical representations •  The relationship between templates and

articulatory skills in P1 and P8 is not as straightforward as with the other subjects

Correlations between the measures (for all subjects except P1 and P8)

r

Template score and PCC-‐R -‐0,94 *, p < 0.05

Template score and variega>on -‐0,90 *, p < 0.05

Template score and PWP -‐0,77 *, p < 0.1

Template score and number of consonants -‐0,46

Answers

1.  Do older childen with cri du chat syndrome use templates that are normally associated with far younger typically developing children? •  YES

2.  Does the template score correlate with other measures? •  YES; negative correlations with some

exceptions.

Answers (cont.)

•  Do older children with cri du chat syndrome use templates that are normally associated with far younger typically developing children? •  YES

•  Does the template score correlate with other measures?

Answers (cont.)

3.  If there are such correlations, how can we interpret them, and what do they tell us about templates?

t in cri du chat syndrome - universitetet i oslo...background: cri du chat syndrome (cdcs) •...

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