Effects of Low-Dose Estrogen Replacement During Childhood on Pubertal Development and Gonadotropin Concentrations in Patients With Turner Syndrome: Results of a Randomized, Double-Blind, Placebo-Controlled Clinical Trial

Quigley, Charmian A.; Wan, Xiaohai; Garg, Sipi; Kowal, Karen; Cutler, Gordon B.; Ross, Judith L.

doi:10.1210/jc.2013-4518

Cited by 55 publications

(42 citation statements)

References 59 publications

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“…The initial dose of EE 2 , 1-5 ng/kg in group E, was extremely low even in comparison with the dosage of 25 ng/kg reported in a previous study [10,11]. Additionally, this is the first study ##, ### A significant difference as described in the text.…”

Section: Discussionmentioning

confidence: 57%

“…It is well known that not only BMD acquisition but also bone maturation is estrogen-dependent [19]. In a recently published report by Quigualy et al [10], where the initial dose of EE 2 was 25 ng/kg, breast development was observed in some of the patients. This pubertal The mean and SD are shown.…”

Section: Discussionmentioning

confidence: 96%

“…In adults, estradiol via transdermal administration was reported to be more beneficial for preventing thrombus; this type of administration, by bypassing the liver (avoiding the first-pass effect) and decreased clotting factors, minimizes the risk of thromboembolic events [24,25]. EE 2 was used for the estrogen treatment of group E in the present study in order to enable comparison with other historical studies where EE 2 was used [10,11,26].…”

Section: Fig 2 Volumetric Bmd (Vbmd) In the 3 Groups (E L And S)mentioning

confidence: 99%

“…Recently, Quigley et al reported in a randomizedcontrol study that low dose ethinyl estradiol administration (EE 2 ; 25 ng/kg/day) starting at the age of 5-8 years improved final height [10,11]. They focused on estrogen replacement therapy in TS, and analyzed only height data without addressing bone mineral density (BMD), which is also estrogen-dependent.…”

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confidence: 99%

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Gradually increasing ethinyl estradiol for Turner syndrome may produce good final height but not ideal BMD

et al. 2017

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Section: Discussionmentioning

confidence: 57%

Section: Discussionmentioning

confidence: 96%

Section: Fig 2 Volumetric Bmd (Vbmd) In the 3 Groups (E L And S)mentioning

confidence: 99%

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confidence: 99%

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Gradually increasing ethinyl estradiol for Turner syndrome may produce good final height but not ideal BMD

et al. 2017

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“…One double-blind, placebo-controlled trial using ultra-low-dose oral ethinylestradiol as a growthpromoting agent during the prepubertal period combined with GH demonstrated a modest synergistic increase in AH, normalization of the timing of thelarche for about one-quarter of the girls, and modest improvements in cognition and memory within specific developmental windows (87,143,144,145).…”

Section: Concomitant Treatment With Childhood Ultra-low-dose Estrogenmentioning

confidence: 99%

Clinical practice guidelines for the care of girls and women with Turner syndrome: proceedings from the 2016 Cincinnati International Turner Syndrome Meeting

Gravholt¹,

Andersen

Conway

et al. 2017

European Journal of Endocrinology

909

1,259

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Turner syndrome affects 25-50 per 100,000 females and can involve multiple organs through all stages of life, necessitating multidisciplinary approach to care. Previous guidelines have highlighted this, but numerous important advances have been noted recently. These advances cover all specialty fields involved in the care of girls and women with TS. This paper is based on an international effort that started with exploratory meetings in 2014 in both Europe and the USA, and culminated with a Consensus Meeting held in Cincinnati, Ohio, USA in July 2016. Prior to this meeting, five groups each addressed important areas in TS care: 1) diagnostic and genetic issues, 2) growth and development during childhood and adolescence, 3) congenital and acquired cardiovascular disease, 4) transition and adult care, and 5) other comorbidities and neurocognitive issues. These groups produced proposals for the present guidelines. Additionally, four pertinent questions were submitted for formal GRADE (Grading of Recommendations, Assessment, Development and Evaluation) evaluation with a separate systematic review of the literature. These four questions related to the efficacy and most optimal treatment of short stature, infertility, hypertension, and hormonal replacement therapy. The guidelines project was initiated by the European Society of Endocrinology and the Pediatric Endocrine Society, in collaboration with the European Society for Paediatric Endocrinology, the Endocrine Society, the European Society of Human Reproduction and Embryology, the American Heart Association, the Society for Endocrinology, and the European Society of Cardiology. The guideline has been formally endorsed by the European Society of Endocrinology, the Pediatric Endocrine Society, the European Society for Paediatric Endocrinology, the European Society of Human Reproduction and Embryology and Summary of recommendationsThe recommendations (R) are worded as recommend (strong recommendation) and suggest (weak recommendation). We formally graded only the evidence underlying recommendations for therapeutic choices. The quality of evidence behind the recommendations is classified as very low (⨁◯◯◯), low (⨁⨁◯◯), moderate (⨁⨁⨁◯) and strong (⨁⨁⨁⨁). See further section 'Summary of methods used for guideline development'. Diagnosis and geneticsR 1.1. We recommend considering a diagnosis of TS in phenotypic females with a karyotype containing one X chromosome and complete or partial absence of the second sex chromosome, associated with one or more typical clinical manifestations of TS (⨁⨁⨁⨁). R 1.2. We recommend against considering a diagnosis of TS in females with one X chromosome and a deletion distal to Xq24 on the other X chromosome, and in women over the age of 50 years with less than 5% 45,X mosaicism (⨁⨁◯◯). R 1.3. We suggest that the new general surveillance management guideline applies to TS patients with any karyotype (⨁⨁◯◯). R 1.4. We recommend to consider testing for Turner syndrome (TS) in a female with typical signs (Table 2) (⨁⨁⨁⨁). R 1.5. We ...

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Growth and growth hormone in Turner syndrome: Looking back, looking ahead

Los

Rosenfeld

2019

American J of Med Genetics Pt C

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Short stature is the most ubiquitous feature of Turner syndrome (TS). Today, many girls with TS are treated with recombinant human growth hormone (GH) to accelerate growth in childhood and to improve adult height. Here, we will review the history of our understanding of growth in TS, reflect on the path of clinical trials ultimately leading to regulatory approval for clinical use of GH, discuss factors associated with growth outcomes and survey the current unanswered questions about growth and GH in TS.

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Effects of Low-Dose Estrogen Replacement During Childhood on Pubertal Development and Gonadotropin Concentrations in Patients With Turner Syndrome: Results of a Randomized, Double-Blind, Placebo-Controlled Clinical Trial

Abstract: In addition to previously reported effects on cognitive measures and GH-mediated height gain, childhood estrogen replacement significantly normalized the onset and tempo of puberty. Childhood low-dose estrogen replacement should be considered for girls with Turner syndrome.

Cited by 55 publications

References 59 publications

Gradually increasing ethinyl estradiol for Turner syndrome may produce good final height but not ideal BMD

Gradually increasing ethinyl estradiol for Turner syndrome may produce good final height but not ideal BMD

Clinical practice guidelines for the care of girls and women with Turner syndrome: proceedings from the 2016 Cincinnati International Turner Syndrome Meeting

Growth and growth hormone in Turner syndrome: Looking back, looking ahead

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