Inhibin B in adolescents and young adults with Turner syndrome
-
Maria Francesca Messina
, Tommaso Aversa
, Giuseppina Salzano , Daria Costanzo , Concetta Sferlazzas , Silvestro Mirabelli , Giuseppina Zirilli und Fortunato Lombardo
Abstract
Objective: Primary gonadal failure may occur in most individuals with Turner syndrome (TS). Since ovaries in TS girls undergo premature apoptosis and cryopreservation of ovarian tissue is now feasible, it would be useful to identify a reliable marker of ovarian reserve in these patients. We planned to evaluate ovarian function in a group of TS patients by measuring both traditional markers and inhibin B and to compare these results with those of a control group.
Study design: We enrolled 23 patients with TS and 17 age-matched healthy girls. The median age of our TS patients was 17.6 years. Three out of the 23 patients (13%) showed spontaneous pubertal development and regular menstrual cycles; the remaining 20 (86.9%) presented with primary amenorrhea.
Results: The median level of inhibin B in the TS patients with primary amenorrhea was 42 pg/mL and did not differ significantly among the different subgroups in relation to karyotype. The median inhibin B level in the control group was significantly higher than in the TS girls with primary amenorrhea (83 vs. 42 pg/mL, p<0.00001). In the three patients with TS and spontaneous menstrual cycles, the inhibin B levels were significantly higher when compared to the values of the TS girls with primary amenorrhea.
Conclusion: TS patients with primary amenorrhea have significantly lower levels of inhibin B than TS girls with spontaneous puberty and healthy controls. Inhibin B does not correlate with follicle-stimulating hormone/luteinizing hormone. If our results are confirmed in further studies, inhibin B could become a first-line screening test for assessing ovarian reserve and a longitudinal marker of the possible decline of ovarian function in TS.
References
1. Gravholt CH. Epidemiological, endocrine and metabolic features in Turner syndrome. Eur J Endocrinol 2004;151:657–87.10.1530/eje.0.1510657Suche in Google Scholar
2. Ranke MB, Saenger P. Turner’s syndrome. Lancet 2001;358:309–14.10.1016/S0140-6736(01)05487-3Suche in Google Scholar
3. Davenport ML. Approach to the patient with Turner syndrome. J Clin Endocrinol Metab 2010;95:1487–95.10.1210/jc.2009-0926Suche in Google Scholar PubMed
4. Messina MF, Zirilli G, Civa R, Rulli I, Salzano G, et al. Neurocognitive profile in Turner’s syndrome is not affected by growth impairment. J Pediatr Endocrinol Metab 2007;20:677–84.10.1515/JPEM.2007.20.6.677Suche in Google Scholar
5. Messina MF, Squadrito G, Valenzise M, Maimone S, Iannelli S, et al. Fibroscan: a new noninvasive method for evaluation of liver dysfunction in Turner syndrome. Eur J Clin Invest 2011;41:183–8.10.1111/j.1365-2362.2010.02397.xSuche in Google Scholar PubMed
6. Pasquino AM, Passeri F, Pucarelli I, Segni M, Municchi G. Spontaneous pubertal development in Turner’s syndrome. Italian Study Group for Turner’s Syndrome. J Clin Endocrinol Metab 1997;82:1810–3.Suche in Google Scholar
7. Hovatta O. Pregnancy in women with Turner’s syndrome. Ann Med 1999;31:106–10.10.3109/07853899908998785Suche in Google Scholar
8. Hreinsson JG, Otala M, Fridström M, Borgström B, Rasmussen C, et al. Follicles are found in the ovaries of adolescent girls with Turner’s syndrome. J Clin Endocrinol Metab 2002;87:3618–23.Suche in Google Scholar
9. Gravholt CH, Naeraa RW, Andersson AM, Christiansen JS, Skakkebaek NE. Inhibin A and B in adolescents and young adults with Turner’s syndrome and no sign of spontaneous puberty. Hum Reprod 2002;17:2049–53.10.1093/humrep/17.8.2049Suche in Google Scholar PubMed
10. Purushothaman R, Lazareva O, Oktay K, Ten S. Markers of ovarian reserve in young girls with Turner’s syndrome. Fertil Steril 2010;94:1557–9.10.1016/j.fertnstert.2009.12.026Suche in Google Scholar PubMed
11. Visser JA, Hokken-Koelega AC, Zandwijken GR, Limacher A, Ranke MB, et al. Anti-Mullerian hormone levels in girls and adolescents with Turner syndrome are related to karyotype, pubertal development and growth hormone treatment. Hum Reprod 2013;28:1899–907.10.1093/humrep/det089Suche in Google Scholar PubMed
12. Groome NP, Illingworth PJ, O’Brien M, Pai R, Rodger FE, et al. Measurement of dimeric inhibin B throughout the human menstrual cycle. J Clin Endorcrinol Metab 1996;81:1401–5.Suche in Google Scholar
13. Weiss L. Additional evidence of gradual loss of germ cells in the pathogenesis of streak ovaries in Turner’s syndrome. J Med Genet 1971;8:540–4.10.1136/jmg.8.4.540Suche in Google Scholar PubMed PubMed Central
14. Reynaud K, Cortvrindt R, Verlinde F, De Schepper J, Bourgain C, et al. Number of ovarian follicles in human fetuses with the 45,X karyotype. Fertil Steril 2004;81:1112–9.10.1016/j.fertnstert.2003.12.011Suche in Google Scholar PubMed
15. Conway GS. Clinical manifestations of genetic defects affecting gonadotropins and their receptors. Clin Endocrinol (Oxf) 1996;45:657–63.10.1046/j.1365-2265.1996.8680879.xSuche in Google Scholar PubMed
16. Bachelot A, Rouxel A, Massin N, Dulon J, Courtillot C, et al; POF-GIS Study Group. Phenotyping and genetic studies of 357 consecutive patients presenting with premature ovarian failure. Eur J Endocrinol 2009;161:179–87.10.1530/EJE-09-0231Suche in Google Scholar PubMed
17. Nardo LG, Christodoulou D, Gould D, Roberts SA, Fitzgerald CT, et al. Anti-Müllerian hormone levels and antral follicle count in women enrolled in vitro fertilization cycles: relationship to lifestyle factors, chronological age and reproductive history. Gynecol Endocrinol 2007;23:486–93.10.1080/09513590701532815Suche in Google Scholar PubMed
18. Sehested A, Juul A, Andersson AM, Petersen JH, Jensen TK, et al. Serum Inibin A and Inibin B in healthy prepubertal, pubertal, and adolescent girls and adult women: relation to age, stage of puberty, menstrual cycle, follicle- stimulating hormone, luteinizing hormone and estradiol levels. J Clin Endocrinol Metab 2000; 85:1634–40.Suche in Google Scholar
19. Hadnott TN, Gould HN, Gharib AM, Bondy CA. Outcomes of spontaneous and assisted pregnancies in Turner syndrome: the U.S. National Institutes of Health experience. Fertil Steril 2011;95:2251–6.10.1016/j.fertnstert.2011.03.085Suche in Google Scholar PubMed PubMed Central
20. Bryman I, Sylvén L, Berntorp K, Innala E, Bergström I, et al. Pregnancy rate and outcome in Swedish women with Turner syndrome. Fertil Steril 2011;95:2507–10.10.1016/j.fertnstert.2010.12.039Suche in Google Scholar PubMed
21. Lockwood GM, Muttukrishna S, Groome NP, Matthews DR, Ledger WL. Mid-follicular phase pulses of inhibin B are absent in polycystic ovarian syndrome and are initiated by successful laparoscopic ovarian diathermy: a possible mechanism regulating emergence of the dominant follicle. J Clin Endocrinol Metab 1998;83:1730–5.10.1210/jcem.83.5.4756Suche in Google Scholar PubMed
22. Hagen CP, Main KM, Kjaergaard S, Juul A. FSH, LH, inhibin B and estradiol levels in Turner syndrome depend on age and karyotype: longitudinal study of 70 Turner girls with or without spontaneous puberty. Hum Reprod 2010;25:3134–41.10.1093/humrep/deq291Suche in Google Scholar PubMed
©2015 by De Gruyter
Artikel in diesem Heft
- Frontmatter
- Highlight: Turner syndrome
- Turner syndrome – working together with patients and their families
- Turner syndrome patients with bicuspid aortic valves and renal malformations exhibit abnormal expression of X-linked inhibitor of apoptosis protein (XIAP)
- Inhibin B in adolescents and young adults with Turner syndrome
- Mode of initial presentation and chromosomal abnormalities in Irish patients with Turner syndrome: a single-centre experience
- Retrospective evaluation of pubertal development and linear growth of girls with Turner Syndrome treated with oral and transdermal estrogen
- Turner syndrome in Albania and the efficacy of its treatment with growth hormone
- Review articles
- The relation of vitamin D status with metabolic syndrome in childhood and adolescence: an update
- Putting the pieces together: cryptorchidism – do we know everything?
- Original articles
- Investigation of androgen receptor gene mutations in a series of 21 patients with 46,XY disorders of sex development
- Molecular diagnosis of maturity-onset diabetes of the young (MODY) in Turkish children by using targeted next-generation sequencing
- Association of physical activity level with depression, anxiety, and quality of life in children with type 1 diabetes mellitus
- Neonatal diabetes in Ukraine: incidence, genetics, clinical phenotype and treatment
- The effect of intraoperative administration of dexamethasone for PONV prophylaxis on perioperative blood glucose level in obese and normal weight children
- Are obesity and metabolic syndrome associated with plasma adropin levels in children?
- Carnitine insufficiency in children with inborn errors of metabolism: prevalence and treatment efficacy
- Comparison of energy expenditure, body composition, metabolic disorders, and energy intake between obese children with a history of craniopharyngioma and children with multifactorial obesity
- Effect of serum cholesterol on bone mineral density in normal-weight children and adolescents
- Assessment of bone turnover markers and bone mineral density in normal short boys
- Long-term treatment for hyperphenylalaninemia and phenylketonuria: a risk for nutritional vitamin B12 deficiency?
- Risk factors affecting the development of nephrocalcinosis, the most common complication of hypophosphatemic rickets
- Children with hyperthyroidism younger than age 7 require higher mg/kg doses of methimazole to normalize free T4 compared to older children
- Effects of bisphosphonates to treat osteoporosis in children with cerebral palsy: a meta-analysis
- AMH levels at central precocious puberty and premature thelarche: is it a parameter?
- Validating Paediatric Morphometrics: body proportion measurement using photogrammetric anthropometry
- Treatment of central precocious puberty and early puberty with GnRH analog in girls with Williams-Beuren syndrome
- Short communication
- Pilot study on the dietary habits and lifestyles of girls with idiopathic precocious puberty from the city of Rome: potential impact of exposure to flame retardant polybrominated diphenyl ethers
- Patient reports
- Hypomagnesemia due to two novel TRPM6 mutations
- A novel mutation of AMH in three siblings with persistent Mullerian duct syndrome
- Kocher-Debré-Semelaigne syndrome with rhabdomyolysis and increased creatinine
- Anaphylaxis to gonadorelin acetate in a girl with central precocious puberty
- Efficacy and safety of sirolimus in a neonate with persistent hypoglycaemia following near-total pancreatectomy for hyperinsulinaemic hypoglycaemia
Artikel in diesem Heft
- Frontmatter
- Highlight: Turner syndrome
- Turner syndrome – working together with patients and their families
- Turner syndrome patients with bicuspid aortic valves and renal malformations exhibit abnormal expression of X-linked inhibitor of apoptosis protein (XIAP)
- Inhibin B in adolescents and young adults with Turner syndrome
- Mode of initial presentation and chromosomal abnormalities in Irish patients with Turner syndrome: a single-centre experience
- Retrospective evaluation of pubertal development and linear growth of girls with Turner Syndrome treated with oral and transdermal estrogen
- Turner syndrome in Albania and the efficacy of its treatment with growth hormone
- Review articles
- The relation of vitamin D status with metabolic syndrome in childhood and adolescence: an update
- Putting the pieces together: cryptorchidism – do we know everything?
- Original articles
- Investigation of androgen receptor gene mutations in a series of 21 patients with 46,XY disorders of sex development
- Molecular diagnosis of maturity-onset diabetes of the young (MODY) in Turkish children by using targeted next-generation sequencing
- Association of physical activity level with depression, anxiety, and quality of life in children with type 1 diabetes mellitus
- Neonatal diabetes in Ukraine: incidence, genetics, clinical phenotype and treatment
- The effect of intraoperative administration of dexamethasone for PONV prophylaxis on perioperative blood glucose level in obese and normal weight children
- Are obesity and metabolic syndrome associated with plasma adropin levels in children?
- Carnitine insufficiency in children with inborn errors of metabolism: prevalence and treatment efficacy
- Comparison of energy expenditure, body composition, metabolic disorders, and energy intake between obese children with a history of craniopharyngioma and children with multifactorial obesity
- Effect of serum cholesterol on bone mineral density in normal-weight children and adolescents
- Assessment of bone turnover markers and bone mineral density in normal short boys
- Long-term treatment for hyperphenylalaninemia and phenylketonuria: a risk for nutritional vitamin B12 deficiency?
- Risk factors affecting the development of nephrocalcinosis, the most common complication of hypophosphatemic rickets
- Children with hyperthyroidism younger than age 7 require higher mg/kg doses of methimazole to normalize free T4 compared to older children
- Effects of bisphosphonates to treat osteoporosis in children with cerebral palsy: a meta-analysis
- AMH levels at central precocious puberty and premature thelarche: is it a parameter?
- Validating Paediatric Morphometrics: body proportion measurement using photogrammetric anthropometry
- Treatment of central precocious puberty and early puberty with GnRH analog in girls with Williams-Beuren syndrome
- Short communication
- Pilot study on the dietary habits and lifestyles of girls with idiopathic precocious puberty from the city of Rome: potential impact of exposure to flame retardant polybrominated diphenyl ethers
- Patient reports
- Hypomagnesemia due to two novel TRPM6 mutations
- A novel mutation of AMH in three siblings with persistent Mullerian duct syndrome
- Kocher-Debré-Semelaigne syndrome with rhabdomyolysis and increased creatinine
- Anaphylaxis to gonadorelin acetate in a girl with central precocious puberty
- Efficacy and safety of sirolimus in a neonate with persistent hypoglycaemia following near-total pancreatectomy for hyperinsulinaemic hypoglycaemia