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Abstract

The thoughtful response of Franzese et al prompts a clarification that is critical to interpreting the results of our 2004 article in the context of the findings of our earlier work and that of others. Although the 12-month data from our ongoing longitudinal study revealed that GH treatment before age 18 months improved mobility skill acquisition, this conclusion was not exclusive of or in contradiction to our previous findings that GH therapy improved the strength and agility skills in older children with PWS.1Carrel A.L. Myers S.E. Whitman B.Y. Allen D.B. Growth hormone improves body composition, fat utilization, physical strength and agility, and growth in Prader-Willi syndrome controlled study.J Pediatr. 1999; 134: 215-221Abstract Full Text Full Text PDF PubMed Scopus (185) Google ScholarWe applaud the pilot work of Franzese et al for their interest in examining the efficacy of a 24-month protocol for GH treatment. They used Touwen’s Test for Children with Minor Neurological Dysfunction to evaluate the neurologic outcome of the 4 boys that they followed during GH treatment. Of concern, however, are studies critiquing the lack of sufficient construct, content, and criterion validity for Touwen’s test.2Kakebeeke T. Jongmans M.J. Dubowitz L.M. Schoemaker M.M. Henderson S.E. Some aspects of the reliability of Touwen’s examination of the child with minor neurological dysfunction.Dev Med Child Neurol. 1993; 35: 1097-1105Crossref PubMed Scopus (25) Google Scholar, 3Wilson P.H. Practitioner review. Approaches to assessment and treatment of children with DCD: an evaluative review.J Child Psychol Psychiatry. 2005; 46: 806-823Crossref PubMed Scopus (176) Google Scholar The problem that challenges us and unites us with Franzese et al is to identify a mechanism of action for GH therapy as it relates to motor outcome. We hold that this will be best facilitated by randomized, controlled studies with sufficient numbers of subjects and psychometrically sound instruments to address the complexity of the motor issues in this population. The thoughtful response of Franzese et al prompts a clarification that is critical to interpreting the results of our 2004 article in the context of the findings of our earlier work and that of others. Although the 12-month data from our ongoing longitudinal study revealed that GH treatment before age 18 months improved mobility skill acquisition, this conclusion was not exclusive of or in contradiction to our previous findings that GH therapy improved the strength and agility skills in older children with PWS.1Carrel A.L. Myers S.E. Whitman B.Y. Allen D.B. Growth hormone improves body composition, fat utilization, physical strength and agility, and growth in Prader-Willi syndrome controlled study.J Pediatr. 1999; 134: 215-221Abstract Full Text Full Text PDF PubMed Scopus (185) Google Scholar We applaud the pilot work of Franzese et al for their interest in examining the efficacy of a 24-month protocol for GH treatment. They used Touwen’s Test for Children with Minor Neurological Dysfunction to evaluate the neurologic outcome of the 4 boys that they followed during GH treatment. Of concern, however, are studies critiquing the lack of sufficient construct, content, and criterion validity for Touwen’s test.2Kakebeeke T. Jongmans M.J. Dubowitz L.M. Schoemaker M.M. Henderson S.E. Some aspects of the reliability of Touwen’s examination of the child with minor neurological dysfunction.Dev Med Child Neurol. 1993; 35: 1097-1105Crossref PubMed Scopus (25) Google Scholar, 3Wilson P.H. Practitioner review. Approaches to assessment and treatment of children with DCD: an evaluative review.J Child Psychol Psychiatry. 2005; 46: 806-823Crossref PubMed Scopus (176) Google Scholar The problem that challenges us and unites us with Franzese et al is to identify a mechanism of action for GH therapy as it relates to motor outcome. We hold that this will be best facilitated by randomized, controlled studies with sufficient numbers of subjects and psychometrically sound instruments to address the complexity of the motor issues in this population. Growth hormone therapy in children with Prader-Willi syndromeThe Journal of PediatricsVol. 148Issue 6PreviewRecently, Carrel et al1 reported that growth hormone (GH) treatment for 12 months improved mobility skill acquisition, especially motor abilities and speech, in infants and toddlers affected by Prader-Willi syndrome (PWS) only when treatment started before 18 months of age. We report on the results obtained in 4 boys with PWS, 4.5 to 9.0 years of age, after GH therapy for 24 months (1 mg/m2 per day, all days) on the intelligence quotient, examined by the WPPSI or Wechsler Intelligence Scale for Children-R tests,2 and the neurological performances, examined by the Touwen test. Full-Text PDF