Adrenal insufficiency in patients with Prader-Willi syndrome

The generalized dysfunction of the hypothalamic-pituitary axis in patients with Prader-Willi syndrome (PWS) is the most likely cause of hypogonadism, inadequate growth hormone secretion, excessive appetite and associated obesity, impaired body temperature regulation, and hypothyroidism. The syndrome...
Ausführliche Beschreibung

Gespeichert in:

Autor*in:	Marcin Jerzy Kusz [verfasserIn] Aneta Monika Gawlik [verfasserIn]

Format:	E-Artikel
Sprache:	Englisch

Erschienen:	2022

Schlagwörter:	PWS Prader-Willi syndrome adrenal insufficiency Synacthen LDSST

Übergeordnetes Werk:	In: Frontiers in Endocrinology - Frontiers Media S.A., 2011, 13(2022)
Übergeordnetes Werk:	volume:13 ; year:2022

Links:	Link aufrufen Link aufrufen Link aufrufen Journal toc

DOI / URN:	10.3389/fendo.2022.1021704

Katalog-ID:	DOAJ083830308

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allfieldsSound	10.3389/fendo.2022.1021704 doi (DE-627)DOAJ083830308 (DE-599)DOAJffe10c99ac09488abb4729c143689266 DE-627 ger DE-627 rakwb eng RC648-665 Marcin Jerzy Kusz verfasserin aut Adrenal insufficiency in patients with Prader-Willi syndrome 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier The generalized dysfunction of the hypothalamic-pituitary axis in patients with Prader-Willi syndrome (PWS) is the most likely cause of hypogonadism, inadequate growth hormone secretion, excessive appetite and associated obesity, impaired body temperature regulation, and hypothyroidism. The syndrome is also related to an increased risk of central adrenal insufficiency, although its prevalence remains unknown. The results of the studies in which different methods of pharmacological stimulation were used do not provide conclusive outcomes. As a result, there are no clear guidelines with regard to diagnosis, prevention, or long-term care when adrenal insufficiency is suspected in patients with PWS. Currently, most patients with PWS are treated with recombinant human growth hormone (rhGH). It has been confirmed that rhGH therapy has a positive effect on growth, body composition, body mass index (BMI), and potentially on psychomotor development in children with PWS. Additionally, rhGH may reduce the conversion of cortisone to cortisol through inhibition of 11β-hydroxysteroid dehydrogenase type 1. However, its influence on basal adrenal function and adrenal stress response remains unexplained in children with PWS. This paper reviews the literature related to the hypothalamic-pituitary-adrenal axis dysfunction in the PWS patient population with a focus on children. PWS Prader-Willi syndrome adrenal insufficiency Synacthen LDSST Diseases of the endocrine glands. Clinical endocrinology Marcin Jerzy Kusz verfasserin aut Aneta Monika Gawlik verfasserin aut In Frontiers in Endocrinology Frontiers Media S.A., 2011 13(2022) (DE-627)645090948 (DE-600)2592084-4 16642392 nnns volume:13 year:2022 https://doi.org/10.3389/fendo.2022.1021704 kostenfrei https://doaj.org/article/ffe10c99ac09488abb4729c143689266 kostenfrei https://www.frontiersin.org/articles/10.3389/fendo.2022.1021704/full kostenfrei https://doaj.org/toc/1664-2392 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022
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callnumber-first	R - Medicine
author	Marcin Jerzy Kusz
spellingShingle	Marcin Jerzy Kusz misc RC648-665 misc PWS misc Prader-Willi syndrome misc adrenal insufficiency misc Synacthen misc LDSST misc Diseases of the endocrine glands. Clinical endocrinology Adrenal insufficiency in patients with Prader-Willi syndrome
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topic_title	RC648-665 Adrenal insufficiency in patients with Prader-Willi syndrome PWS Prader-Willi syndrome adrenal insufficiency Synacthen LDSST
topic	misc RC648-665 misc PWS misc Prader-Willi syndrome misc adrenal insufficiency misc Synacthen misc LDSST misc Diseases of the endocrine glands. Clinical endocrinology
topic_unstemmed	misc RC648-665 misc PWS misc Prader-Willi syndrome misc adrenal insufficiency misc Synacthen misc LDSST misc Diseases of the endocrine glands. Clinical endocrinology
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title	Adrenal insufficiency in patients with Prader-Willi syndrome
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title_full	Adrenal insufficiency in patients with Prader-Willi syndrome
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title_sort	adrenal insufficiency in patients with prader-willi syndrome
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title_auth	Adrenal insufficiency in patients with Prader-Willi syndrome
abstract	The generalized dysfunction of the hypothalamic-pituitary axis in patients with Prader-Willi syndrome (PWS) is the most likely cause of hypogonadism, inadequate growth hormone secretion, excessive appetite and associated obesity, impaired body temperature regulation, and hypothyroidism. The syndrome is also related to an increased risk of central adrenal insufficiency, although its prevalence remains unknown. The results of the studies in which different methods of pharmacological stimulation were used do not provide conclusive outcomes. As a result, there are no clear guidelines with regard to diagnosis, prevention, or long-term care when adrenal insufficiency is suspected in patients with PWS. Currently, most patients with PWS are treated with recombinant human growth hormone (rhGH). It has been confirmed that rhGH therapy has a positive effect on growth, body composition, body mass index (BMI), and potentially on psychomotor development in children with PWS. Additionally, rhGH may reduce the conversion of cortisone to cortisol through inhibition of 11β-hydroxysteroid dehydrogenase type 1. However, its influence on basal adrenal function and adrenal stress response remains unexplained in children with PWS. This paper reviews the literature related to the hypothalamic-pituitary-adrenal axis dysfunction in the PWS patient population with a focus on children.
abstractGer	The generalized dysfunction of the hypothalamic-pituitary axis in patients with Prader-Willi syndrome (PWS) is the most likely cause of hypogonadism, inadequate growth hormone secretion, excessive appetite and associated obesity, impaired body temperature regulation, and hypothyroidism. The syndrome is also related to an increased risk of central adrenal insufficiency, although its prevalence remains unknown. The results of the studies in which different methods of pharmacological stimulation were used do not provide conclusive outcomes. As a result, there are no clear guidelines with regard to diagnosis, prevention, or long-term care when adrenal insufficiency is suspected in patients with PWS. Currently, most patients with PWS are treated with recombinant human growth hormone (rhGH). It has been confirmed that rhGH therapy has a positive effect on growth, body composition, body mass index (BMI), and potentially on psychomotor development in children with PWS. Additionally, rhGH may reduce the conversion of cortisone to cortisol through inhibition of 11β-hydroxysteroid dehydrogenase type 1. However, its influence on basal adrenal function and adrenal stress response remains unexplained in children with PWS. This paper reviews the literature related to the hypothalamic-pituitary-adrenal axis dysfunction in the PWS patient population with a focus on children.
abstract_unstemmed	The generalized dysfunction of the hypothalamic-pituitary axis in patients with Prader-Willi syndrome (PWS) is the most likely cause of hypogonadism, inadequate growth hormone secretion, excessive appetite and associated obesity, impaired body temperature regulation, and hypothyroidism. The syndrome is also related to an increased risk of central adrenal insufficiency, although its prevalence remains unknown. The results of the studies in which different methods of pharmacological stimulation were used do not provide conclusive outcomes. As a result, there are no clear guidelines with regard to diagnosis, prevention, or long-term care when adrenal insufficiency is suspected in patients with PWS. Currently, most patients with PWS are treated with recombinant human growth hormone (rhGH). It has been confirmed that rhGH therapy has a positive effect on growth, body composition, body mass index (BMI), and potentially on psychomotor development in children with PWS. Additionally, rhGH may reduce the conversion of cortisone to cortisol through inhibition of 11β-hydroxysteroid dehydrogenase type 1. However, its influence on basal adrenal function and adrenal stress response remains unexplained in children with PWS. This paper reviews the literature related to the hypothalamic-pituitary-adrenal axis dysfunction in the PWS patient population with a focus on children.
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title_short	Adrenal insufficiency in patients with Prader-Willi syndrome
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Adrenal insufficiency in patients with Prader-Willi syndrome

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