Literature DB >> 32378142

From mini-puberty to pre-puberty: early impairment of the hypothalamus-pituitary-gonadal axis with normal testicular function in children with non-mosaic Klinefelter syndrome.

M Spaziani1, S Granato2, N Liberati3, F M Rossi2, N Tahani2, C Pozza2, D Gianfrilli2, G Papi2, A Anzuini2, A Lenzi2, L Tarani3, A F Radicioni2.   

Abstract

PURPOSE: Klinefelter syndrome (KS) is a genetic disorder caused by the presence of an extra X chromosome in males. The aim of this study was to evaluate the hypothalamic-pituitary-gonadal (HPG) axis and the clinical profile of KS boys from mini-puberty to early childhood. PATIENTS AND METHODS: In this retrospective, cross-sectional, population study, 145 KS boys and 97 controls aged 0-11.9 years were recruited. Serum FSH, LH, testosterone (T), Inhibin B (INHB), sex hormone binding globulin (SHBG) and anti-Müllerian hormone (AMH) were determined. Auxological parameters were assessed. To better represent the hormonal and clinical changes that appear in childhood, the entire population was divided into 3 groups: ≤ 6 months (group 1; mini-puberty); > 6 months and ≤ 8 years (group 2; early childhood); > 8 and ≤ 12 years (group 3; mid childhood).
RESULTS: During mini-puberty (group 1), FSH and LH were significantly higher in KS infants than controls (p < 0.05), as were INHB and T (respectively p < 0.0001 and p < 0.005). INHB was also significantly higher in KS than controls in group 2 (p < 0.05). AMH appeared higher in KS than in controls in all groups, but the difference was only statistically significant in group 2 (p < 0.05). No significant differences were found in height, weight, testicular volume, and penile length.
CONCLUSIONS: No hormonal signs of tubular or interstitial damage were found in KS infants. The presence of higher levels of gonadotropins, INHB and testosterone during mini-puberty and pre-puberty may be interpreted as an alteration of the HPG axis in KS infants.

Entities:  

Keywords:  Gonadal axis (HPG); Hypothalamic; Infancy; Klinefelter syndrome; Leydig cells; Mini-puberty; Pituitary; Sertoli cells

Year:  2020        PMID: 32378142     DOI: 10.1007/s40618-020-01281-x

Source DB:  PubMed          Journal:  J Endocrinol Invest        ISSN: 0391-4097            Impact factor:   4.256


  41 in total

1.  Prenatal and postnatal prevalence of Klinefelter syndrome: a national registry study.

Authors:  Anders Bojesen; Svend Juul; Claus Højbjerg Gravholt
Journal:  J Clin Endocrinol Metab       Date:  2003-02       Impact factor: 5.958

Review 2.  Strategies and advantages of early diagnosis in Klinefelter's syndrome.

Authors:  A F Radicioni; E De Marco; D Gianfrilli; S Granato; L Gandini; A M Isidori; A Lenzi
Journal:  Mol Hum Reprod       Date:  2010-04-14       Impact factor: 4.025

3.  Is the prevalence of Klinefelter syndrome increasing?

Authors:  Joan K Morris; Eva Alberman; Claire Scott; Patricia Jacobs
Journal:  Eur J Hum Genet       Date:  2007-11-14       Impact factor: 4.246

Review 4.  Consensus statement on diagnosis and clinical management of Klinefelter syndrome.

Authors:  A F Radicioni; A Ferlin; G Balercia; D Pasquali; L Vignozzi; M Maggi; C Foresta; A Lenzi
Journal:  J Endocrinol Invest       Date:  2010-12       Impact factor: 4.256

Review 5.  Klinefelter syndrome, insulin resistance, metabolic syndrome, and diabetes: review of literature and clinical perspectives.

Authors:  Andrea Salzano; Roberta D'Assante; Liam M Heaney; Federica Monaco; Giuseppe Rengo; Pietro Valente; Daniela Pasquali; Eduardo Bossone; Daniele Gianfrilli; Andrea Lenzi; Antonio Cittadini; Alberto M Marra; Raffaele Napoli
Journal:  Endocrine       Date:  2018-03-23       Impact factor: 3.633

6.  47,XXY (Klinefelter syndrome) and 47,XYY: estimated rates of and indication for postnatal diagnosis with implications for prenatal counselling.

Authors:  L Abramsky; J Chapple
Journal:  Prenat Diagn       Date:  1997-04       Impact factor: 3.050

Review 7.  Klinefelter syndrome: cardiovascular abnormalities and metabolic disorders.

Authors:  A E Calogero; V A Giagulli; L M Mongioì; V Triggiani; A F Radicioni; E A Jannini; D Pasquali
Journal:  J Endocrinol Invest       Date:  2017-03-03       Impact factor: 4.256

Review 8.  Klinefelter's syndrome.

Authors:  Fabio Lanfranco; Axel Kamischke; Michael Zitzmann; Eberhard Nieschlag
Journal:  Lancet       Date:  2004 Jul 17-23       Impact factor: 79.321

Review 9.  Klinefelter syndrome: more than hypogonadism.

Authors:  George A Kanakis; Eberhard Nieschlag
Journal:  Metabolism       Date:  2018-01-31       Impact factor: 8.694

Review 10.  Klinefelter syndrome (KS): genetics, clinical phenotype and hypogonadism.

Authors:  M Bonomi; V Rochira; D Pasquali; G Balercia; E A Jannini; A Ferlin
Journal:  J Endocrinol Invest       Date:  2016-09-19       Impact factor: 4.256

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  2 in total

1.  Adverse pathophysiological influence of early testosterone therapy on the testes of boys with higher grade sex chromosome aneuploidies (HGAs): a retrospective, cross-sectional study.

Authors:  M Spaziani; C Tarantino; C Pozza; A Anzuini; F Panimolle; G Papi; D Gianfrilli; A Lenzi; A F Radicioni
Journal:  J Endocrinol Invest       Date:  2020-10-24       Impact factor: 4.256

Review 2.  The role of scrotal ultrasonography from infancy to puberty.

Authors:  Matteo Spaziani; Claudio Lecis; Chiara Tarantino; Emilia Sbardella; Carlotta Pozza; Daniele Gianfrilli
Journal:  Andrology       Date:  2021-06-11       Impact factor: 3.842

  2 in total

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