title: Insulin-like growth factor I improves height in growth hormone insensitivity: two years' results.
abstract: Thirty-one patients with growth hormone insensitivity syndrome (GHIS) and 2 with GH gene deletion (age 11.2 (3.7-22.9) years; BA (GP) 8.2 years; height -6.5 +/- 1.6 SDS) were recruited for the multicenter study. At birth, length was more retarded (-1.38 SDS) than weight (-0.56 SDS). The rhIGF-I dose was 40-120 micrograms/kg BW twice daily s.c. In 26 patients, first year HV increased from 3.9 +/- 1.8 to 8.5 +/- 2.1 cm/year (delta (d) HT SDS 0.8 +/- 0.5). In 18 patients, second year HV was 6.4 +/- 2.2 cm/year (dHT SDS 0.4 +/- 0.5). There was normal progression of puberty. Mean progression of BA was 1.2 and 1.5 years/year during the first and second year. There was no dose effect of IGF-I on growth. Weight-for-height index (WHI) and skinfold thickness were significantly correlated at start, 12 and 24 months (r = 0.83, 0.87 and 0.79). Changes in WHI were positively correlated with dHT SDS during the first and second year (r = 0.54, 0.56). Serum IGF-I rose, IGF-II decreased, and IGFBP-3 remained constant. Adverse events were (number of occasions): headache (21) (early); hypoglycemia (13); papilloedema (1) (reversible); Bell's palsy (1) (reversible); lipohypertrophy (7) (late); tonsillectomy/adenoidectomy (3) (late). The results show that there is effective long-term treatment of GHIS with systemically administered IGF-I and support the view that IGFBPs play an important role in the action of IGF-I.
authors.affiliation: University Children's Hospitals, Tübingen, Germany.
authors.forename: M B
authors.initials: MB
authors.lastname: Ranke
authors.forename: M O
authors.initials: MO
authors.lastname: Savage
authors.forename: P G
authors.initials: PG
authors.lastname: Chatelain
authors.forename: M A
authors.initials: MA
authors.lastname: Preece
authors.forename: R G
authors.initials: RG
authors.lastname: Rosenfeld
authors.forename: W F
authors.initials: WF
authors.lastname: Blum
authors.forename: P
authors.initials: P
authors.lastname: Wilton
mesh_terms: D000293:Adolescent; D000328:Adult; D000375:Aging / Q000502:physiology; D001823:Body Composition / Q000187:drug effects; D002648:Child; D002675:Child, Preschool; D005260:Female; D006130:Growth Disorders / Q000188:drug therapy* / Q000503:physiopathology; D013006:Growth Hormone / Q000502:physiology*; D006801:Humans; D007334:Insulin-Like Growth Factor I / Q000009:adverse effects / Q000378:metabolism / Q000627:therapeutic use*; D007335:Insulin-Like Growth Factor II / Q000378:metabolism; D008297:Male; D013997:Time Factors
chemical_list: D007334:Insulin-Like Growth Factor I; D007335:Insulin-Like Growth Factor II; D013006:Growth Hormone