Congenital idiopathic growth hormone deficiency associated with prenatal and early postnatal growth failure

Gluckman, Peter D.; Gunn, Alistair J.; Wray, Alison; Cutfield, Wayne S.; Chatelain, Pierre; Guilbaud, Olivier; Ambler, Geoffrey; Wilton, Patrick; Albertsson‐Wikland, Kerstin

doi:10.1016/s0022-3476(05)80342-7

Cited by 163 publications

(87 citation statements)

References 13 publications

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“…Similarly, Gluckman et al (26) reported in 52 GHD patients an increasing frequency of height Ն¹2 SDS with age: 21% at birth, 70% at 6 months and 91% at 12 months. In our patients as well as in other reports Table 3 Current auxological data (means Ϯ S.D.…”

Section: Discussionmentioning

confidence: 70%

Long-term results of GH therapy in GH-deficient children treated before 1 year of age

Huet

Jc²,

Nivelon³

et al. 1999

European Journal of Endocrinology

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Objectives: To evaluate the long-term effects of GH therapy in early diagnosed GH-deficient patients treated before 1 year of age. Study design: We studied all 59 patients (33 males) recorded by Association France-Hypophyse and treated with GH (0.50 Ϯ 0.15 IU/kg (S.D.) per week) before 1 year of age. Clinical presentation and growth parameters under GH treatment were analyzed. Results: Neonatal manifestations of hypopituitarism were frequent: hypoglycemia (n ¼ 50), jaundice (n ¼ 25) and micropenis (n ¼ 17/33). Although birth length was moderately reduced (¹0.9 Ϯ 1.4), growth retardation at diagnosis (5.8 Ϯ 3.8 months) was severe (¹3.5 Ϯ 1.9 standard deviation scores (SDS)). Fifty patients (85%) had thyrotropin and/or corticotropin deficiency. After a mean duration of GH therapy of 8.0 Ϯ 3.6 years, change in height SDS was þ3:11 Ϯ 2:06 S.D., exceeding 4 SDS in 19 patients. Only 9 patients (15%) did not reach a height of ¹2 S.D. for chronological age and 20 patients (34%) exceeded their target height. Pretreatment height SDS was independently associated with total catch-up growth. Conclusion: Conventional doses of GH allow normalization of height in patients with early GH deficiency and treatment.

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Section: Discussionmentioning

confidence: 70%

Long-term results of GH therapy in GH-deficient children treated before 1 year of age

Huet

Jc²,

Nivelon³

et al. 1999

European Journal of Endocrinology

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“…One might speculate on a non-GH-dependent mechanism: as ghrelin and GHSR mRNAs have been demonstrated in pancreatic islets and ghrelin stimulates insulin secretion (23), GHSR variants may enhance ghrelin-mediated insulin release promoting fetal growth. On the other hand, reduced birth length has also been noted in GH-deficient babies, suggesting a possible minor influence of GH on prenatal growth (24). To our knowledge, rs482204 and rs562416 have not been studied before in relation to stature.…”

Section: Discussionmentioning

confidence: 96%

GH secretagogue receptor gene polymorphisms are associated with stature throughout childhood

Riedl

Hughes²,

Harris³

et al. 2012

European Journal of Endocrinology

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Context: Ghrelin plays a major role in GH physiology and energy metabolism. Polymorphisms of its receptor (GH secretagogue receptor (GHSR)) may influence childhood growth and weight regulation. Objective: To correlate GHSR polymorphisms with auxological parameters throughout childhood in a healthy cohort. Study design: Longitudinal retrospective population-based genetic association study. Subjects and methods: GHSR genotypes were evaluated in 1362 children and compared with height/length, weight, and body mass index (BMI) data across an observation span of 10 years (0, 1, 3, 5, 8, and 10 years). Five different GHSR SNPs (rs2922126, rs2981464, rs482204, rs562416, and rs572169), minor allele frequency O0.1, were genotyped. Identification of potential genetic associations with height, weight, and BMI, using additive and dominant/recessive models, was optimized by comparing allele or genotype frequencies between the tallest and the shortest 27% of subjects for each auxological variable. Significance of association was evaluated by c 2 test. Results: The rs482204 TT genotype, vs TC/CC, was associated with greater stature across the entire observation period (P!0.05). Similarly, the rs562416 TT genotype, vs TG/GG, correlated positively with tall stature at 3, 8, and 10 years. Other SNPs and genotypes showed no association with height at any age. No association was found between any tested SNPs and weight or BMI. Conclusions: Longitudinal investigation between birth and 10 years in a population-based cohort revealed a significant association of the rs482204 and rs562416 GHSR polymorphisms on height, whereas no association between GHSR polymorphisms and weight or BMI was ascertainable.

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“…Insulin-like growth factor (IGF)-I has been shown to be a major growth factor during fetal life and umbilical cord blood IGF-I concentrations are positively correlated with size at birth (Gluckman et al 1992b). IGF-I infusion beyond an experimentally induced oesophageal atresia in fetal sheep, calculated to replace the amount of IGF-I otherwise swallowed, prevented somatic and gastrointestinal growth restriction seen in saline-treated fetuses (Kimble et al 1999).…”

Section: Introductionmentioning

confidence: 99%

Chronic pulsatile infusion of growth hormone to growth-restricted fetal sheep increases circulating fetal insulin-like growth factor-I levels but not fetal growth

Bauer

Breier²,

Bloomfield³

et al. 2003

Journal of Endocrinology

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Intra-uterine growth restriction (IUGR) is a major cause of perinatal mortality and morbidity. Postnatally, growth hormone (GH) increases growth, increases circulating insulin-like growth factor (IGF)-I levels, and alters metabolism. Our aim was to determine if GH infusion to IUGR fetal sheep would alter fetal growth and metabolism, and thus provide a potential intra-uterine treatment for the IUGR fetus.We studied three groups of fetuses: control, IUGR+ vehicle and IUGR+GH (n=5 all groups). IUGR was induced by repeated embolisation of the placental vascular bed between 110 and 116 days of gestation (term=145 days). GH (3·5 mg/kg/day) or vehicle was infused in a pulsatile manner from 117 to 127 days of gestation.Embolisation reduced fetal growth rate by 25% (P<0·01) and reduced the weight of the fetal liver (20%), kidney (23%) and thymus (31%; all P<0·05). GH treatment further reduced the weight of the fetal kidneys (32%) and small intestine (35%; both P<0·04), but restored the relative weight of the fetal thymus and liver (P<0·05). Embolisation decreased fetal plasma IGF-I concentrations (48%, P<0·001) and increased IGF binding protein 1 (IGFBP-1) concentrations (737%, P<0·002). GH treatment restored fetal plasma IGF-I concentrations to control levels, while levels in IUGR+vehicle fetuses stayed low (P<0·05 vs control). IGFBP-1 and IGFBP-2 concentrations were about sevenfold lower in amniotic fluid than in fetal plasma, but amniotic and plasma concentrations were closely correlated (r=0·75, P<0·0001 and r=0·55 P<0·0001 respectively). Embolisation transiently decreased fetal blood oxygen content (40%, P<0·002), and increased blood lactate concentrations (213%, P<0·04). Both returned to pre-embolisation levels after embolisation stopped, but blood glucose concentrations declined steadily in IUGR+vehicle fetuses. GH treatment maintained fetal blood glucose concentrations at control levels.Our study shows that GH infusion to the IUGR fetal sheep restores fetal IGF-I levels but does not improve fetal growth, and further reduces the fetal kidney and intestine weights. Thus, fetal GH therapy does not seem a promising treatment stratagem for the IUGR fetus.

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Congenital idiopathic growth hormone deficiency associated with prenatal and early postnatal growth failure

Cited by 163 publications

References 13 publications

Long-term results of GH therapy in GH-deficient children treated before 1 year of age

Long-term results of GH therapy in GH-deficient children treated before 1 year of age

GH secretagogue receptor gene polymorphisms are associated with stature throughout childhood

Chronic pulsatile infusion of growth hormone to growth-restricted fetal sheep increases circulating fetal insulin-like growth factor-I levels but not fetal growth

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