Literature DB >> 21347587

Cross-sectional analysis of speech and cognitive performance in 32 patients with classic galactosemia.

Björn Hoffmann1, Udo Wendel, Susanne Schweitzer-Krantz.   

Abstract

BACKGROUND: Long-term outcome in classic galactosemia is disappointing with impaired IQ, reduced bone mineral density, and fertility problems. Moreover, speech impairment is common with conflicting reports regarding frequency, pattern, and relation to IQ.
OBJECTIVE: To evaluate speech and cognitive performance in patients with galactosemia.
METHODS: Speech performance was evaluated by means of the Hierarchische Wortlisten, a German word-repetition test for the diagnosis of apraxia of speech, using real words and pseudo-words. Cognitive performance was evaluated by use of age-appropriate German versions of the Wechsler Scales.
RESULTS: In a cohort of 32 patients (12 females, 20 males; mean age 21.2 ± 7.2 years) with classic galactosemia, the mean IQ was 76.2 ± 14.8. Eighty-four percent of the patients passed the speech test with errors. Speech errors were much more related to pseudo-words than real words and were predominantly observed in words with three and four syllables. The performance in producing words was correlated to the IQ scores.
CONCLUSION: Impairment of speech affects a significant number of patients with galactosemia, appears in early childhood, and persists into adulthood. The pattern of speech impairment may allow labeling as apraxia of speech. In many cases impaired speech is related to decreased IQ.

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Year:  2011        PMID: 21347587     DOI: 10.1007/s10545-011-9297-5

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  28 in total

1.  Verbal dyspraxia in children with galactosemia.

Authors:  D Nelson
Journal:  Eur J Pediatr       Date:  1995       Impact factor: 3.183

2.  Ovarian failure in galactosaemia.

Authors:  F Kaufman; M D Kogut; G N Donnell; H Koch; U Goebelsmann
Journal:  Lancet       Date:  1979-10-06       Impact factor: 79.321

3.  Developmental apraxia of speech: I. Descriptive and theoretical perspectives.

Authors:  L D Shriberg; D M Aram; J Kwiatkowski
Journal:  J Speech Lang Hear Res       Date:  1997-04       Impact factor: 2.297

4.  Neuropsychological and linguistic follow-up studies of children with galactosaemia from an unscreened population.

Authors:  T W Hansen; B Henrichsen; R K Rasmussen; A Carling; A B Andressen; O Skjeldal
Journal:  Acta Paediatr       Date:  1996-10       Impact factor: 2.299

5.  Neurologic complications in galactosemia.

Authors:  T K Koch; K A Schmidt; J E Wagstaff; W G Ng; S Packman
Journal:  Pediatr Neurol       Date:  1992 May-Jun       Impact factor: 3.372

6.  A longitudinal study of cognitive functioning in patients with classical galactosaemia, including a cohort treated with oral uridine.

Authors:  F R Manis; L B Cohn; C McBride-Chang; J A Wolff; F R Kaufman
Journal:  J Inherit Metab Dis       Date:  1997-08       Impact factor: 4.982

7.  Intelligence and quality of dietary treatment in phenylketonuria.

Authors:  I Smith; M G Beasley; A E Ades
Journal:  Arch Dis Child       Date:  1990-05       Impact factor: 3.791

8.  Bone metabolism in galactosemia.

Authors:  B Panis; P Ph Forget; M J P G van Kroonenburgh; C Vermeer; P P Menheere; F H Nieman; M E Rubio-Gozalbo
Journal:  Bone       Date:  2004-10       Impact factor: 4.398

9.  Correlation of cognitive, neurologic, and ovarian outcome with the Q188R mutation of the galactose-1-phosphate uridyltransferase gene.

Authors:  F R Kaufman; J K Reichardt; W G Ng; Y K Xu; F R Manis; C McBride-Chang; J A Wolff
Journal:  J Pediatr       Date:  1994-08       Impact factor: 4.406

10.  Developmental aspects of galactosemia from infancy to childhood.

Authors:  K Fishler; R Koch; G N Donnell; E Wenz
Journal:  Clin Pediatr (Phila)       Date:  1980-01       Impact factor: 1.168

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  7 in total

1.  Living situation, occupation and health-related quality of life in adult patients with classic galactosemia.

Authors:  Björn Hoffmann; Nico Dragano; Susanne Schweitzer-Krantz
Journal:  J Inherit Metab Dis       Date:  2012-03-24       Impact factor: 4.982

Review 2.  Appropriateness of newborn screening for classic galactosaemia: a systematic review.

Authors:  L Varela-Lema; L Paz-Valinas; G Atienza-Merino; R Zubizarreta-Alberdi; R Vizoso Villares; M López-García
Journal:  J Inherit Metab Dis       Date:  2016-04-26       Impact factor: 4.982

3.  Bone Health in Classic Galactosemia: Systematic Review and Meta-Analysis.

Authors:  Britt van Erven; Lindsey Welling; Sandra C van Calcar; Artemis Doulgeraki; François Eyskens; Joanna Gribben; Eileen P Treacy; Rein Vos; Susan E Waisbren; M Estela Rubio-Gozalbo; Annet M Bosch
Journal:  JIMD Rep       Date:  2016-12-20

4.  Systematic Review and Meta-analysis of Intelligence Quotient in Early-Treated Individuals with Classical Galactosemia.

Authors:  Lindsey Welling; Susan E Waisbren; Kevin M Antshel; Hugh-Owen Colhoun; Matthias Gautschi; Stephanie Grünewald; Rebecca Holman; Johanna H van der Lee; Eileen P Treacy; Annet M Bosch
Journal:  JIMD Rep       Date:  2017-04-09

5.  Cryptic residual GALT activity is a potential modifier of scholastic outcome in school age children with classic galactosemia.

Authors:  Emily L Ryan; Mary Ellen Lynch; Elles Taddeo; Tyler J Gleason; Michael P Epstein; Judith L Fridovich-Keil
Journal:  J Inherit Metab Dis       Date:  2013-01-15       Impact factor: 4.982

Review 6.  Fertility preservation in female classic galactosemia patients.

Authors:  Britt van Erven; Cynthia S Gubbels; Ron J van Golde; Gerard A Dunselman; Josien G Derhaag; Guido de Wert; Joep P Geraedts; Annet M Bosch; Eileen P Treacy; Corrine K Welt; Gerard T Berry; M Estela Rubio-Gozalbo
Journal:  Orphanet J Rare Dis       Date:  2013-07-16       Impact factor: 4.123

7.  Cognitive functioning in patients with classical galactosemia: a systematic review.

Authors:  Merel E Hermans; Mendy M Welsink-Karssies; Annet M Bosch; Kim J Oostrom; Gert J Geurtsen
Journal:  Orphanet J Rare Dis       Date:  2019-10-18       Impact factor: 4.123

  7 in total

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