Abnormal vascular and neural retinal morphology in congenital lifetime isolated growth hormone deficiency

Pereira-Gurgel, Virginia M.; Faro, Augusto C N; Salvatori, Roberto; Chagas, Thiago A.; Carvalho-Junior, José F.; Oliveira, Carla R. P.; Costa, Úrsula; Melo, Gustavo Barreto; Hellström, Ann; Aguiar‐Oliveira, Manuel H.

doi:10.1016/j.ghir.2016.07.001

Cited by 14 publications

(14 citation statements)

References 16 publications

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“…They also present a mild retinal phenotype, with a moderate reduction in vascular retinal branching points associated to an increase in optic disc size, but with similar thickness of the macula (40), as also observed in LS (24). Therefore, an increase in optic disc size in a child with severe short stature may suggest congenital IGHD (31).…”

Section: Overall Homozygous Phenotypementioning

confidence: 74%

“…These features have been published in 42 different publications (2,19,22,25,27,29,30,31,32,33,36,37,38,40,41,44,45,46,47,48,49,50,51,52,53,54,56,57,61,62,63,64,66,67,68,74,75,76,77,78,79,80). We have used this experiment of nature to discover 'usual laws of nature' as suggested by William Harvey, and to understand some aspects of more common conditions such as short stature, insulin resistance, diabetes, atherosclerosis, osteoporosis, cancer and ultimately longevity.…”

Section: Introductionmentioning

confidence: 99%

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MECHANISMS IN ENDOCRINOLOGY: The multiple facets of GHRH/GH/IGF-I axis: lessons from lifetime, untreated, isolated GH deficiency due to a GHRH receptor gene mutation

Aguiar‐Oliveira

Souza

Oliveira

et al. 2017

European Journal of Endocrinology

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Twenty years ago, we described kindred of 105 individuals with isolated GH deficiency (IGHD) in Itabaianinha County, in northeast Brazil, carrying a homozygous mutation in the GH-releasing hormone receptor gene. These subjects exhibit markedly reduced GH responsiveness to stimulatory tests, and anterior pituitary hypoplasia. Serum concentrations of IGF-I, IGF binding protein type 3 and the acid-labile subunit are markedly reduced, with a lesser reduction of IGF-II. The most striking physical findings of these IGHD individuals are the proportionate short stature, doll facies, high-pitched voice and visceral obesity with reduced fat-free mass. There is neither microphallus, nor neonatal hypoglycemia. Puberty is delayed, menopause anticipated, but fertility is preserved in both genders. The reduction in bone sizes is not even, with mean standard deviation scores for height of −7.2, total maxillary length of −6.5, total facial height of −4.3 and cephalic perimeter of −2.7. In addition, the non-osseous growth is not uniform, preserving some organs, like pancreas, liver, kidney, brain and eyes, and compromising others such as thyroid, heart, uterus and spleen. These subjects present higher prevalence of dizziness, mild high-tones sensorineural hearing loss, reduction of vascular retinal branching points, increase of optic disk, genu valgum and increased systolic blood pressure. Biochemically, they have high low density lipoprotein cholesterol and C-reactive protein levels, but maintain increased insulin sensitivity, and do not show premature atherosclerosis. Finally, they have normal immune function, and normal longevity. This review details the findings and summarizes 20 years of clinical research carried out in this unique population.

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Section: Overall Homozygous Phenotypementioning

confidence: 74%

Section: Introductionmentioning

confidence: 99%

MECHANISMS IN ENDOCRINOLOGY: The multiple facets of GHRH/GH/IGF-I axis: lessons from lifetime, untreated, isolated GH deficiency due to a GHRH receptor gene mutation

Aguiar‐Oliveira

Souza

Oliveira

et al. 2017

European Journal of Endocrinology

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“…Consequently, most of the previous studies relevant with IGHD were focused on the retina. 4,8,24 Normal macular thickness and decreased retinal fiber layer thickness were found to be associated with IGHD according to these studies.…”

Section: Discussionmentioning

confidence: 56%

“…After birth, little or no vascularization occurs and most of the retinal vascularization happens during fetal period. 24 This process is regulated by several local produced growth factors as a response to hypoxia. Hormones and factors in circulation except GH alone affect the production and action of these local factors.…”

Section: Discussionmentioning

confidence: 99%

Choroidal thickness measurements in children with isolated growth hormone deficiency

Yalçın

Aktaş

Yüce

et al. 2017

Eye

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PurposeThe aim of this study was to determine the choroidal thickness measurement values in cases with isolated growth hormone deficiency (IGHD), to compare them with the healthy control group by using enhanced depth imaging optical coherence tomography (EDI-OCT), and to evaluate the effect of growth hormone (GH) treatment on choroid.Patients and methodsIn this study, 23 cases who were diagnosed with IGHD as a study group and 46 healthy subjects as a control group were included. All patients and controls underwent a complete ophthalmologic examination, including an examination with EDI-OCT. Choroidal thickness (CT) was measured at the fovea and at 1000 μm intervals from the foveal center in both temporal and nasal directions.ResultsThe mean subfoveal choroidal thickness (SFCT) was 329.04±88.49 μm in the cases with IGHD and 365.35±50.48 μm in the control group (P=0.033). The mean CT at temporal 1 and 2 mm were thinner in the IGHD group than that of control group (P=0.033 and P=0.043, respectively). Nasal quadrant measurements were also found to be thinner in the IGHD cases than that of control group, but the difference was not statistically significant. We found a significant positive correlation between pubertal staging and SFCT (r=0.607, P=0.006). There was no statistically significant difference in CT values of the study group between before and 12 months after GH treatment (P>0.05).ConclusionThis study shows patients with IGHD has a thinner CT when compared with healthy pediatric cases. GH treatment seems to be not associated with the choroidal development.

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“…These authors also drew attention to the importance of GH and IGF-1 for normal retinal vascularization. More recently, Pereira-Gurgel et al ( 13 ) reported moderate reduction of retinal vascular branching points in isolated GHD patients. Another recent study also evaluated the effects of the GH/IGF-1 axis on retinal vascular branching and other characteristics in patients with GH insensitivity and reported reduced retinal vasculature and tortuosity of the retinal vessels ( 14 ).…”

Section: Discussionmentioning

confidence: 96%

Retinal Neural and Vascular Structure in Isolated Growth Hormone Deficiency Children and Evaluation of Growth Hormone Treatment Effect

Yüce

Yalçın

Bıdecı

et al. 2018

Jcrpe

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Objective:To evaluate neural and vascular retinal morphology of children with isolated growth hormone deficiency (GHD) and to determine any retinal changes due to GH treatment.Methods:Twenty-eight children with isolated GHD and 53 age-, gender- and body mass index-matched healthy volunteers were enrolled in this prospective study. The retinal nerve fibre layer (RNFL) and macular thickness (MT) were measured, as well as intraocular pressure (IOP). The number of retinal vascular branching points were calculated. Effect of GH treatment on the retina and IOP was evaluated after one year of treatment. Measurements were also made in the control group at baseline and following the initial examination. Pre- and post-treatment changes were compared. The findings were also compared with those of the controls. The correlation between ocular dimensions and insulin-like growth factor-I (IGF-1) levels were also analysed.Results: The number of branching points was significantly lower in GHD patients as compared with control subjects (15.11±2.67 and 19.70±3.37, respectively, p=0.05 for all comparisons). No statistically significant differences were found in mean RNFL, MT and IOP values between GHD patients and control subjects. GH treatment did not create any significant changes in the retinal vascularization or other retinal neural parameters and IOP either within the patient group or when compared with the control group. No correlations were observed between ocular dimensions and IGF-1 levels.Conclusion:Our findings suggest that isolated GHD may lead to decreased retinal vascularization. However, retinal neural growth and differentiation were not affected by GHD. These findings may be related to the fetal development process of pituitary somatotropic cells and the retina. Additionally, GH treatment did not cause any changes in retinal neural and vascular tissues.

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Abnormal vascular and neural retinal morphology in congenital lifetime isolated growth hormone deficiency

Cited by 14 publications

References 16 publications

MECHANISMS IN ENDOCRINOLOGY: The multiple facets of GHRH/GH/IGF-I axis: lessons from lifetime, untreated, isolated GH deficiency due to a GHRH receptor gene mutation

MECHANISMS IN ENDOCRINOLOGY: The multiple facets of GHRH/GH/IGF-I axis: lessons from lifetime, untreated, isolated GH deficiency due to a GHRH receptor gene mutation

Choroidal thickness measurements in children with isolated growth hormone deficiency

Retinal Neural and Vascular Structure in Isolated Growth Hormone Deficiency Children and Evaluation of Growth Hormone Treatment Effect

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