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Prader-Labhart-Willi syndrome: auxological response to a conventional dose of growth hormone in patients with classical growth hormone deficiency.
European Journal of Medical Research 2000 July 20
The aim of the study was to evaluate the effect of growth hormone (GH) (14 IU/m2/week) on height, body mass index and predicted adult height in children with Prader-Labhart-Willi (PLWS) syndrome and GH deficiency. - By auxological criteria GH deficiency was suspected in 10 patients (age: 7-16 years). In 9 patients two GH provocative testings and MR imaging of the pituitary were performed. IGF-I measurements, bone age determinations, body mass index (BMI), height standard deviation score, height velocity -SDS and predicted adult height (PAH) were determined before and during GH treatment. Nine patients could be followed for up to 2 years, 3 patients for 4 years and 2 patients for 5 years of GH treatment. - Predicted adult height could be calculated in all after 1 year of treatment and was on average 11.6 cm below target height (-17.6 to +2.5 cm). GH treatment in a conventional dose (14 IU/m2/week) in daily subcutaneous injections was performed. Height SDS improved from -3.47 to -2.27 after 2 years of treatment, height velocity SDS from -1.74 to +2.65 after 1 year and remained +1.78 after 2 years. PAH increased on average by 5.1 cm. BMI was unchanged during 2 years. Bone age development was appropriate with +2.13 years after 2 years. In 2 patients who were treated with GH for up to 5 years and in 1 who was treated up to 4 years, the positive effect on height SDS and height velocity SDS persisted. - In conclusion GH treatment in a conventional dose (14 IU/m2/week) in PLWS patients with GH deficiency is effective over a period of 2 years in regard to height development and predicted adult height but ony limited in regard to changes in BMI.
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