Therapeutic Efficacy and Safety of GH in Japanese Children with Down Syndrome Short Stature Accompanied by GH Deficiency

Meguri, Kyoko; Inoue, Masato; Narahara, K; Sato, Takahiro; Takata, Ami; Ohki, Nobuhiko; Ozono, Keiichi

doi:10.1297/cpe.22.65

Cited by 4 publications

(5 citation statements)

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“…Several reports have found similar differences highlighting short stature as a typical feature in DS, and growth velocity being markedly reduced, mainly between the ages ranging from six months to three years (27). Deficiencies in the secretion of growth hormone (GH) which regulates the production of Insulin-like growth factor (IGFs) have been clearly associated to the marked reduction in speed of growth in young children with DS (28, 29), and consistently with those findings, it has been demonstrated that this phenotype can be normalized with GH therapy (30, 31).…”

Section: Discussionmentioning

confidence: 88%

Profiling of circulating chromosome 21-encoded microRNAs, miR-155 and Let-7c, in Down Syndrome People

Perez-Villareal

Aviña-Padilla

Lopez

et al. 2020

Preprint

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Down syndrome (DS), or Trisomy 21 (Ts21), is the most common chromosomal survival aneuploidy. Nevertheless, people with DS have compromised health, and the increase in their life expectancy further heightens the risk of developing chronic degenerative diseases such as obesity, dyslipidemias and diabetes mellitus associated with higher morbidity, and mortality for cardiovascular disease from an early age. DS is also accompanied by a higher risk of neurodegeneration. The extra genetic material that characterizes DS causes an imbalance in the genetic dosage, including overexpression of miR-155 and Let-7c miRNAs, both associated with cognitive impairment and dementia in adults. The dynamics of expression of their putative target genes in the early stages of the development of DS and their clinical associations, however, remain to be ascertained. This study aimed to evaluate the relative expression of miR-155 and Let-7c in young and adult individuals with DS and its possible association with biochemical indicators of lipid metabolism. The anthropometric, clinical, biochemical, and gene expression features of miR-155 and Let-7c were analyzed in a population of 52 control and 50 DS subjects divided into groups of 20 years of age or younger and 21 years or older. Expression changes for miR-155 were not significant. Nevertheless, a negative correlation for HDL-Cholesterol concentrations and miR-155 expression was identified. Notably, Let-7c was overexpressed in DS from young and old ages. Overall, our results suggest that Let-7c is related from early stages to cognitive impairment in DS, while a similar role of miR-155 in late stages could be mediated by alterations in lipid metabolism. Further studies with both miRNAs will shed light on their potential as therapeutic targets to prevent or delay cognitive impairment in DS.

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Section: Discussionmentioning

confidence: 88%

Profiling of circulating chromosome 21-encoded microRNAs, miR-155 and Let-7c, in Down Syndrome People

Perez-Villareal

Aviña-Padilla

Lopez

et al. 2020

Preprint

View full text Add to dashboard Cite

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“…Patients with DS also often show impaired stature that can be linked to a GH deficit (6,19) and can be corrected, at least in part, with the hormone replacement therapy. In subjects that also have CD, the response to the therapy is less satisfactory.…”

Section: Discussionmentioning

confidence: 99%

Corneal Alterations in Subjects with Downs Syndrome and Celiac Disease

Bubella¹

2018

Arch Ophthalmol Optom

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Aim: To assess the presence of changes in corneal thickness in. the patients with Down's syndrome (DS) and celiac disease (CD)Materials and Methods: Fortyeight subjects with Down's syndrome ( 23 men and 24 women) were enrolled in the study.22 subjects were suffered from celiac disease too .Results: 4 subjects ( 8.5%) , including 3 with CD, presented keratoconus at stage 1 or 2 according to the Amsler-Krumeich classification.The pachymetric examination in subjects with celiac disease showed in 12( 54,5%) a corneal thickness at the lower limit of the relevant range for the respective age groups. and in 10 (45,5) a pathological thickness.In subjects with only DS the corneal thickness was at the lower limit of the relevant range in 18(72%) Conclusions: Subjects with DS and celiac disease too present a reduced corneal thickness and a higher frequency of keratoconus.

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“…Another study reported on mild subclinical hypothyroidism (19). Precocious puberty was observed in one boy 3.4 yrs after GH treatment initiation (24). In no study was the development of leukemia reported.…”

Section: Adverse Effectsmentioning

confidence: 98%

“…No GH deficiency diagnosis was required in three of the four studies included in the mini meta-analysis (21)(22)(23). A diagnosis of GH deficiency was required in one study included in the meta-analysis (24), as well as in two other studies that could not be included (19,20).…”

Section: Height Outcomementioning

confidence: 99%

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GH treatment in pediatric Down syndrome: a systematic review and mini meta-analysis

et al. 2023

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ObjectiveTo analyze and determine the safety and efficacy of growth hormone (GH) treatment in Down syndrome (DS) pediatric patients and to weigh ethical aspects involved.DesignSystematic review and mini meta-analysis of the literature.MethodsA search was performed in PubMed, Embase, Scopus, and PsycINFO through August 2022. Eligible studies included those who answered at least one of the following two questions: 1) What is the effect of growth hormone treatment in children with Down syndrome? 2) What are the ethical arguments in favor and against growth hormone treatment for children with Down syndrome? Multiple reviewers independently screened each article for eligibility.ResultsIn total sixteen reports detailed medical effects of GH treatment in pediatric DS patients and eight studies dealt with ethical aspects of GH treatment. Treatment with GH resulted in significantly higher growth velocity in patients with DS. The ethical complexity is great but does not present insurmountable difficulties to the therapeutic option.ConclusionsAs GH treatment is safe and effective for short-term height growth, GH therapy should be considered in long-term treatment of DS children.

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Therapeutic Efficacy and Safety of GH in Japanese Children with Down Syndrome Short Stature Accompanied by GH Deficiency

Cited by 4 publications

References 4 publications

Profiling of circulating chromosome 21-encoded microRNAs, miR-155 and Let-7c, in Down Syndrome People

Profiling of circulating chromosome 21-encoded microRNAs, miR-155 and Let-7c, in Down Syndrome People

Corneal Alterations in Subjects with Downs Syndrome and Celiac Disease

GH treatment in pediatric Down syndrome: a systematic review and mini meta-analysis

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