Impact of short stature on quality of life: A systematic literature review

IF 1.6 4区 医学 Q4 CELL BIOLOGY
Philippe Backeljauw , Marco Cappa , Wieland Kiess , Lisa Law , Charlotte Cookson , Caroline Sert , John Whalen , Mehul T. Dattani
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引用次数: 26

Abstract

Objective

We sought to obtain a better understanding of the burden of short stature using a systematic literature review.

Methods

Studies of the burden of short stature, of any cause in adults and children, were searched using Embase, MEDLINE and Cochrane databases in April 2020, capturing publications from 2008 onwards. Case series and populations with adult-onset growth hormone deficiency (GHD) were excluded.

Results

Of 1684 publications identified, 41 studies (33 in children, 8 in adults) were included. All studies assessed human burden. Most study populations in children included short stature due to GHD, idiopathic short stature (ISS) and short stature after being born small for gestational age (SGA). In these populations, four studies showed that quality of life (QoL) in children with short stature was significantly worse than in children with normal stature. A significant association between QoL and short stature was observed in children with chronic kidney disease (CKD) (3 studies), achondroplasia (1 study) and transfusion-dependent β-thalassaemia (1 study), and in samples with mixed causes of short stature (3 studies). Three studies (one in GHD/ISS/SGA and two in CKD) found no significant association between short stature and QoL, and several studies did not report statistical significance. Approximately half of adult studies showed that QoL was reduced with short stature, and the other half showed no association. Two studies, one in adults with Prader–Willi syndrome and one in children with GHD, suggested a potential association between short stature and poorer cognitive outcomes. Three studies demonstrated an increased caregiver burden in parents of children with short stature.

Conclusions

Evidence suggests that, compared with those with normal stature, children and adults with short stature of any cause may experience poorer QoL. Further research could extend our understanding of the human burden in this field.

身材矮小对生活质量的影响:系统的文献综述
目的通过系统的文献回顾,我们试图更好地了解矮小的负担。方法于2020年4月使用Embase、MEDLINE和Cochrane数据库检索成人和儿童中任何原因的矮小负担的研究,获取2008年以来的出版物。排除了成年性生长激素缺乏症(GHD)的病例系列和人群。结果在1684篇出版物中,纳入了41项研究(33项儿童研究,8项成人研究)。所有研究都评估了人类负担。大多数儿童研究人群包括由于GHD导致的身材矮小,特发性身材矮小(ISS)和出生时小于胎龄(SGA)后身材矮小。在这些人群中,四项研究表明,身材矮小的儿童的生活质量(QoL)明显低于身材正常的儿童。在患有慢性肾脏疾病(CKD)(3项研究)、软骨发育不全(1项研究)和输血依赖性β-地中海贫血(1项研究)的儿童以及混合原因导致的身材矮小(3项研究)中,观察到生活质量与身材矮小之间的显著关联。三项研究(一项针对GHD/ISS/SGA,两项针对CKD)发现身材矮小与生活质量之间无显著关联,有几项研究未报告统计学意义。大约一半的成人研究表明,生活质量随着身材矮小而降低,而另一半则没有发现任何关联。两项研究,一项针对患有普瑞德-威利综合症的成年人,另一项针对患有GHD的儿童,都表明身材矮小和认知能力差之间存在潜在联系。三项研究表明,矮小儿童的父母照顾负担增加。结论有证据表明,与身高正常的人相比,任何原因导致的身高不足的儿童和成人的生活质量都较差。进一步的研究可以扩展我们对这一领域人类负担的理解。
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来源期刊
Growth Hormone & Igf Research
Growth Hormone & Igf Research 医学-内分泌学与代谢
CiteScore
3.30
自引率
0.00%
发文量
38
审稿时长
57 days
期刊介绍: Growth Hormone & IGF Research is a forum for research on the regulation of growth and metabolism in humans, animals, tissues and cells. It publishes articles on all aspects of growth-promoting and growth-inhibiting hormones and factors, with particular emphasis on insulin-like growth factors (IGFs) and growth hormone. This reflects the increasing importance of growth hormone and IGFs in clinical medicine and in the treatment of diseases.
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