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lüll The essential role of IGF-I: lessons from the long-term study and treatment of children and adults with Laron syndrome Laron ZJ Clin Endocrinol Metab 1999[Dec]; 84 (12): 4397-404Fifty patients with primary GH resistance (Laron syndrome) due to molecular defects of the GH receptor or post-receptor pathways were followed from infancy through adulthood. This condition leading to long-term insulin-like growth factor-I (IGF-I) deprivation caused marked growth retardation (-4 to 8 height SD), acromicia, organomicria, retarded development of the skeletal and muscular systems, a small cranium, slow motor development, and impairment of intellectual development in some of the patients. In addition, there was progressive obesity, insulin resistance, a tendency for hypoglycemia, followed later in life by hypercholesterolemia and by glucose intolerance and even diabetes. IGF-I treatment of children with Laron syndrome, by our and other groups (150-240 microg/day sc), stimulated growth (8 cm in the first year and 4-5 cm in the following years) and normalized the biochemical abnormalities. Overdosage led to adverse effects such as hypoglycemia, edema, swelling of soft tissues, and hyperandrogenism. It is concluded that primary IGF-I deprivation induces severe auxological, biochemical, and hormonal changes, the only treatment being biosynthetic IGF-I administration.|*Drug Resistance[MESH]|*Human Growth Hormone[MESH]|Adolescent[MESH]|Adult[MESH]|Child[MESH]|Child, Preschool[MESH]|Female[MESH]|Humans[MESH]|Insulin-Like Growth Factor I/*deficiency/*physiology/therapeutic use[MESH]|Male[MESH]|Mutation[MESH]|Receptors, Somatotropin/genetics[MESH]|Recombinant Proteins/therapeutic use[MESH] |