9
views
0
recommends
+1 Recommend
0 collections
    0
    shares
      • Record: found
      • Abstract: found
      • Article: found
      Is Open Access

      Recurrent maternal virilization during pregnancy in patients with PCOS: two clinical cases

      brief-report

      Read this article at

      Bookmark
          There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

          Abstract

          Background

          Maternal virilization during pregnancy is a rare phenomenon. Polycystic ovary syndrome (PCOS), luteoma and luteinic cysts are the most frequent and benign etiologies. This article presents two cases of recurrent maternal virilization during pregnancy.

          Clinical cases

          Our reported cases were young women with Afro-Caribbean and Nigerian origins. Data were collected by history-taking, clinical examination, laboratory investigations, transabdominal ultrasonographic examination and Magnetic Resonance Imaging. Both patients were diagnosed with PCOS according to the Rotterdam criteria. During each of their pregnancies they both developed an explosive hirsutism, a deepening in the voice, a clitoromegaly. Gestational diabetes occurred during pregnancies. There was no fetal virilization, despite raising androgen levels, more than tenfold to normal. Improvement of hirsutism and normalization of androgens were described in postpartum.

          Conclusion

          Only few cases of maternal virilization during pregnancy were reported in literature and even fewer concern recurrent and bilateral ovarian etiology. Hyperplasia of ovarian theca cells seems to be the most likely explanation, which would suggest that PCOS belongs to a spectrum of abnormal reactivity of the ovary to human Chorionic Gonadotrophin (hCG) stimulation along with luteoma and luteinic cyst of pregnancy.  Insulin resistance could worsen hyperandrogenism but is not enough to explain virilization. Treatment should focus on protecting the fetus of possible virilization as well as its mother, but also on preserving the subsequent fertility in both.

          Related collections

          Most cited references28

          • Record: found
          • Abstract: found
          • Article: not found

          Elevated prenatal anti-Müllerian hormone reprograms the fetus and induces polycystic ovary syndrome in adulthood

          Polycystic ovary syndrome (PCOS) is the main cause of female infertility worldwide and corresponds with a high degree of comorbidities and economic burden. How PCOS is passed on from one generation to the next is not clear, but it may be a developmental condition. Most women with PCOS exhibit higher levels of circulating luteinizing hormone, suggestive of heightened gonadotropin-releasing hormone (GnRH) release, and Anti-Müllerian Hormone (AMH) as compared to healthy women. Excess AMH in utero may affect the development of the female fetus. However, as AMH levels drop during pregnancy in women with normal fertility it was unclear if their levels were also elevated in pregnant women with PCOS. Here, we measured AMH in a cohort of pregnant women with PCOS and control women and found that AMH is significantly more elevated in the former group versus the latter. To determine if the elevation of AMH during pregnancy in women with PCOS is a bystander effect or a driver of the condition in the offspring, we modelled our clinical findings by treating pregnant mice with AMH and followed the neuroendocrine phenotype of their female progeny postnatally. This treatment resulted in maternal neuroendocrine-driven testosterone excess and diminished placental metabolism of testosterone to estradiol, resulting in a masculinization of the exposed female fetus and a PCOS-like reproductive and neuroendocrine phenotype in adulthood. We found that the affected females had persistently hyperactivated GnRH neurons and that GnRH antagonist treatment in the adult female offspring restored their neuroendocrine phenotype to a normal state. These findings highlight a critical role for excess prenatal AMH exposure and subsequent aberrant GnRH receptor signaling in the neuroendocrine dysfunctions of PCOS, while offering a new potential therapeutic avenue to treat the condition during adulthood.
            Bookmark
            • Record: found
            • Abstract: found
            • Article: not found

            Serum anti-Mullerian hormone as a surrogate for antral follicle count for definition of the polycystic ovary syndrome.

            Despite its frequency, the polycystic ovary syndrome (PCOS) is still a difficult diagnosis in endocrinology, gynecology, and reproductive medicine. To help solve this issue, the Rotterdam consensus conference proposed to include the ultrasonographic follicle count as a new diagnostic criterion, in addition to hyperandrogenism and oligo-anovulation. Unfortunately, its assessment does not offer sufficient reliability worldwide. The aim of our study was to check whether anti-Müllerian hormone (AMH) measurement in the serum could be a surrogate for antral follicle count in the diagnostic criteria of PCOS. Serum AMH was measured with a second-generation immunoassay in a cohort of 73 PCOS patients and 96 controls, and its diagnostic power was evaluated by receiver operating characteristic curves. PCOS was diagnosed according to the Rotterdam definition. Serum AMH levels were 3-fold higher in PCOS patients than in controls (81.6 vs. 33.5 pmol/liter; P < 0.001) and were significantly related to the follicle number in the two groups. The area under the receiver operating characteristic curve for the AMH assay was 0.851, indicating a good diagnostic potency. Setting the threshold at 60 pmol/liter offered the best compromise between specificity (92%) and sensitivity (67%). The serum AMH level is an accurate marker of the ovarian early antral follicle number and offers a good diagnostic potency. In situations where accurate ultrasonographic data are not available, AMH could thus be used instead of the follicle count as a diagnostic criterion and incorporated as such in the Rotterdam definition of PCOS.
              Bookmark
              • Record: found
              • Abstract: found
              • Article: not found

              Is foetal hyperexposure to androgens a cause of PCOS?

              Polycystic ovary syndrome (PCOS) is the most common endocrinopathy affecting reproductive-aged women. The pathophysiology of this syndrome is still not completely understood but recent evidence suggests that the intra-uterine environment may be a key factor in the pathogenesis of PCOS, in particular, hyperexposure of the foetus to androgens. High concentrations of maternal serum testosterone during pregnancy have been shown to influence behaviour during childhood, the prevalence of autism disorders and anti-Mullerian hormone (AMH) concentrations in adolescence. They are also thought to re-programme the female reproductive axis to induce the features of PCOS in later life: oligo/anovulation, polycystic ovaries, hyperandrogenism and insulin resistance (IR). Support for this developmental theory for the aetiology of PCOS is gathering momentum, following results from first animal studies and now human data, which lend credence to many aspects of this hypothesis.
                Bookmark

                Author and article information

                Contributors
                mariedek@hotmail.com
                agathe.dumont@chru-lille.fr
                aude.bruyneel@chru-lille.fr
                didier.dewailly@chru-lille.fr
                sophie.jonard@chru-lille.fr
                Journal
                Reprod Biol Endocrinol
                Reprod. Biol. Endocrinol
                Reproductive Biology and Endocrinology : RB&E
                BioMed Central (London )
                1477-7827
                30 October 2018
                30 October 2018
                2018
                : 16
                : 107
                Affiliations
                [1 ]ISNI 0000 0004 0593 6676, GRID grid.414184.c, Service de Gynécologie Endocrinienne et Médecine de la Reproduction, Hôpital Jeanne de Flandre, CHRU Lille, ; 2 Avenue Eugène Avinée, 59037 Lille, France
                [2 ]ISNI 0000 0004 0594 3884, GRID grid.418052.a, Centre Hospitalier de Tourcoing, ; 155 rue du Président René Coty, 59200 Tourcoing, France
                [3 ]ISNI 0000 0004 0471 8845, GRID grid.410463.4, Centre Hospitalier Régional Universitaire de Lille, ; 2 Avenue Oscar Lambret, 59037 Lille, France
                Author information
                http://orcid.org/0000-0001-5992-4126
                Article
                428
                10.1186/s12958-018-0428-9
                6208071
                30376853
                2827cc9e-5b26-40f3-89a0-efc2f246a32b
                © The Author(s). 2018

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 10 August 2018
                : 17 October 2018
                Categories
                Short Communication
                Custom metadata
                © The Author(s) 2018

                Human biology
                polycystic ovary syndrome,luteinic cyst,luteoma,pregnancy,virilization,hyperandrogenism,gestational diabetes,insulin resistance

                Comments

                Comment on this article