Mental and motor development before and during growth hormone treatment in infants and toddlers with Prader–Willi syndrome

Festen, Dederieke; Wevers, Maaike; Lindgren, Ann Christin; Böhm, Birgitta; Otten, Barto J.; Wit, Jan M.; Duivenvoorden, Hugo J.; Hokken-Koelega, Anita

doi:10.1111/j.1365-2265.2007.03126.x

Cited by 129 publications

(131 citation statements)

References 31 publications

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“…The improvement was especially noticeable in the more severely hypotonic children, which was consistent with other studies reporting that those with initially lower motor development improved the most with GHT [12,13]. This is important as muscle strength in PWS is correlated with motor performance and muscle weakness is a limiting factor for motor development [38].…”

Section: Accepted Manuscriptsupporting

confidence: 79%

“…There was a concomitant decrease in PWS specific BMI SDS which may have been facilitated by either direct or indirect effects of GHT through improved mobility and physical activity and hence increased energy expenditure [12,13], or through improvements in current dietary advice, stimulation of activity and early intervention in general.…”

Section: Discussionmentioning

confidence: 99%

“…Bakker et al [34] reported that the bioactivity of IGF-I was high in children (age ≤ 10 years) with high IGF-I SDS but less so in older children on GHT, necessitating downward adjustments in GH The recommended starting dose in Australia (4.5 mg/m 2 /week) is substantially lower than those used in other studies [12,13] or presently recommended elsewhere [20], but substantially higher than the dose of 2.1 mg/m 2 /week, which was considered ineffective in the only dose comparison study in PWS to date [21]. In this context it is noteworthy that surface area based dosing (using m 2 ) as opposed to weight based dosing (using kg) results in relatively higher growth hormone doses in infants and younger children than older children [35].…”

Section: Accepted Manuscriptmentioning

confidence: 99%

“…In young children a positive effect on motor function and muscle strength/tone has been noted [10,12]. Developmental milestones may be reached at an earlier age and mental development and adaptive skills may be improved [13,14]. However, in most GH studies reported to date very few children were less than 1 year old when GHT was started and were grouped together in the analysis with toddlers [14,15] or 3 to 4 year old children [16,17].…”

Section: Accepted Manuscript Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Low dose growth hormone treatment in infants and toddlers with Prader-Willi syndrome is comparable to higher dosage regimens

Scheermeyer

Harris

Hughes

et al. 2017

Growth Hormone & IGF Research

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Section: Accepted Manuscriptsupporting

confidence: 79%

Section: Discussionmentioning

confidence: 99%

Section: Accepted Manuscriptmentioning

confidence: 99%

Section: Accepted Manuscript Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Low dose growth hormone treatment in infants and toddlers with Prader-Willi syndrome is comparable to higher dosage regimens

Scheermeyer

Harris

Hughes

et al. 2017

Growth Hormone & IGF Research

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“…3,5 In infancy, severe hypotonia combined with muscle weakness leads to serious motor developmental delay. [5][6][7][8][9] These motor problems persist, although they are less marked, in childhood and adulthood. [10][11][12][13][14][15][16] It is presumed that the motor problems are related to an increased fat:muscle ratio even in underweight infants with PWS.…”

mentioning

confidence: 99%

Growth Hormone Therapy, Muscle Thickness, and Motor Development in Prader-Willi Syndrome: An RCT

Reus

Pillen

Pelzer³

et al. 2014

Pediatrics

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WHAT'S KNOWN ON THIS SUBJECT: Infants with Prader-Willi syndrome suffer from hypotonia, muscle weakness, and motor developmental delay and have increased fat mass combined with decreased muscle mass. Growth hormone improves body composition and motor development.WHAT THIS STUDY ADDS: Ultrasound scans confirmed decreased muscle thickness in infants with Prader-Willi syndrome, which improved as result of growth hormone treatment. Muscle thickness was correlated to muscle strength and motor performance. Catch-up growth in muscle thickness was related to muscle use independent of growth hormone. abstract OBJECTIVE: To investigate the effect of physical training combined with growth hormone (GH) on muscle thickness and its relationship with muscle strength and motor development in infants with Prader-Willi syndrome (PWS). METHODS:In a randomized controlled trial, 22 infants with PWS (12.9 6 7.1 months) were followed over 2 years to compare a treatment group (n = 10) with a waiting-list control group (n = 12). Muscle thickness of 4 muscle groups was measured by using ultrasound. Muscle strength was evaluated by using the Infant Muscle Strength meter. Motor performance was measured with the Gross Motor Function Measurement. Analyses of variance were used to evaluate between-group effects of GH on muscle thickness at 6 months and to compare pre-and posttreatment (after 12 months of GH) values. Multilevel analyses were used to evaluate effects of GH on muscle thickness over time, and multilevel bivariate analyses were used to test relationships between muscle thickness, muscle strength, and motor performance. RESULTS:A significant positive effect of GH on muscle thickness (P , .05) was found. Positive relationships were found between muscle thickness and muscle strength (r = 0.61, P , .001), muscle thickness and motor performance (r = 0.81, P , .001), and muscle strength and motor performance (r = 0.76, P , .001).CONCLUSIONS: GH increased muscle thickness, which was related to muscle strength and motor development in infants with PWS. Catch-up growth was faster in muscles that are most frequently used in early development. Because this effect was independent of GH, it suggests a training effect.

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Outcomes of Adenotonsillectomy in Patients With Prader-Willi Syndrome

Meyer¹,

Splaingard²,

Repaske³

et al. 2012

Arch Otolaryngol Head Neck Surg

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Mental and motor development before and during growth hormone treatment in infants and toddlers with Prader–Willi syndrome

Abstract: One year of GH treatment significantly improved mental and motor development in PWS infants compared to randomized controls.

Cited by 129 publications

References 31 publications

Low dose growth hormone treatment in infants and toddlers with Prader-Willi syndrome is comparable to higher dosage regimens

Low dose growth hormone treatment in infants and toddlers with Prader-Willi syndrome is comparable to higher dosage regimens

Growth Hormone Therapy, Muscle Thickness, and Motor Development in Prader-Willi Syndrome: An RCT

Outcomes of Adenotonsillectomy in Patients With Prader-Willi Syndrome

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