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case report

DOI: https://doi.org/10.5114/pm.2018.78560 Menopause Rev 2018; 17(3): 139-140

Introduction

Premature ovarian insufficiency (POI) affects ap- proximately 1% of women under the age of 40 years and is associated with female infertility [1]. The symp- toms are similar to those of menopause: oligo/amen- orrhea, hypoestrogenism, elevated level of gonado- tropins, and diminished number of follicles within the ovaries. Although the causes of POI are largely un- known, genetic defects such as Turner syndrome and fragile X syndrome premutation, environmental factors, and autoimmune diseases seem to play an important role in the aetiopathology.

Primary ovarian insufficiency is generally diagnosed as part of the evaluation for frequent gynaecological complaints, such as irregular menses, so the diagnosis usually comes unexpectedly. Besides causing infertili- ty, POI is associated with multiple health risks such as menopausal symptoms, decreased bone mineral densi- ty and increased risk of fractures, greater risk of cardio- vascular diseases, and psychological impact, which may include depression and potential early decline in cogni- tion. Ovarian failure is not permanent, which differenti- ates this condition form menopause. Women with POI ovulate extremely rarely. However, 5% of such women conceive spontaneously and have a normal pregnancy after the diagnosis is established [2].

Spontaneous pregnancy in a patient with premature ovarian insufficiency – case report

Anna Calik-Ksepka, Monika Grymowicz, Weronika Bronkiewicz, Aleksandra Urban, Kamil Mierzejewski, Ewa Rudnicka, Roman Smolarczyk

Department of Gynaecological Endocrinology, Medical University of Warsaw, Warsaw, Poland

Abstract

Diagnosis of premature ovarian insufficiency is usually sudden and distressful for the patient in terms of facing infertility. However, some patients with POI have intermittent ovarian activity with an overall 5% proba- bility of pregnancy. We report the case of 27-year-old woman with premature ovarian insufficiency treated with hormone replacement therapy, who six months after diagnosis conceived spontaneously. Apart from bleeding episodes in the first trimester, the pregnancy course was uneventful, and a  healthy neonate was delivered.

Women with premature ovarian insufficiency should be informed about the small but nonetheless real possi- bility of pregnancy.

Key words: premature ovarian insufficiency, spontaneous pregnancy.

Case report

A 27-year-old woman was referred to the gynaeco- logical endocrinology clinic with oligomenorrhoea, and periodic stomach and spine pain. Menarche occurred at age of 16 years, followed by regular menstrual bleed- ing. At 19 years, she commenced on birth control pills that she continued for three years. Her personal history for previous surgery or other relevant pathologies was negative. Laboratory tests showed elevated follicle- stimulating hormone (FSH) level at 102.2 mIU/ml (normal levels 3.03-8.08 mIU/ml), oestradiol (E2) at

< 10 pg/ml (21-251 pg/ml), and luteinising hormone (LH) 45.9 mIU/ml (1.8-11.78 mIU/ml). Repeated labora- tory results confirmed the diagnosis of premature ovar- ian insufficiency. Anti-Müllerian hormone (AMH) was at the very low level of 0.01 ng/ml. Thyroid-stimulating hormone (TSH), prolactin, and androgen status were within normal limits. Lower pelvic transvaginal ultraso- nography found no pathology. The patient’s karyotype corresponded to the normal female karyotype.

Hormone replacement therapy (HRT) was imple- mented to minimise bone loss and decrease the risk of cardiovascular events. The patient was treated with cyclical hormone therapy using oestrogen (2 mg oes- tradiol) with the addition with progestin for 14 days in the cycle (10 mg dydrogesterone). Our patient sponta-

Corresponding author:

Monika Grymowicz, Department of Gynaecological Endocrinology, Medical University of Warsaw, Karowa 2, 00-315 Warsaw, Poland, e-mail: monika.grymowicz@wp.pl

Submitted: 27.06.2018 Accepted: 27.07.2018

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Menopause Review/Przegląd Menopauzalny 17(3) 2018

140

neously and unexpectedly conceived after six months on HRT. After recognising the pregnancy, HRT treatment was discontinued. During the first trimester of pregnan- cy several episodes of vaginal bleeding occurred. Pro- gesterone therapy was then implemented. Pregnancy proceeded without any further complications. At the 39th week of gestation a healthy child was born.

Conclusions

Diagnosis of POI is usually sudden and very dis- tressing for the patient. Due to a  diminished number of follicles, the patients face the prospect of infertility.

Oocyte donation is the only proven and recommended treatment for infertility in women with POI. Cumulative pregnancy rates of oocyte donation treatment are very high (70-80%). However, such treatment is not avail- able in some countries, and not all patients would ac- cept oocyte donation.

However, while normal menopause is generally an irreversible event, POI is characterised by intermittent ovarian function with oestrogen production and even ovulation, despite the presence of high gonadotropin levels. Spontaneous ovulations were observed in 20-24%, and conceptions in about 5%, of women with POI [3, 4].

It is important to admit the probability of ovulation oc- currence in the case of patients with oligo/amenorrhoea.

The chances of resuming ovarian function depend chiefly on whether the amenorrhoea is primary or secondary.

Large studies have shown that numerous mechanisms responsible for the primary amenorrhoea are more ad- vanced and serious than in the case of secondary amen- orrhoea [4, 5]. Unexpectedly, the aetiologies responsible for familial forms of POI may show an increased proba- bility of resuming ovarian function [4].

Another important predictor of resumption of ovar- ian activity are the results of laboratory tests of FSH, oestradiol, and inhibin B levels [4, 5]. AMH levels prove not to be predictive of resumption of ovarian activity in POI women [5]. It is crucial to underline the importance of educating and reassuring patients that spontaneous pregnancies in women with POI are not associated with higher obstetric morbidity or neonatal risk as compared with the general population, and may, just like in the presented case, lead to the birth of a healthy child. It is also important to inform patients who do not want to get pregnant about the necessity of contraception usage, because of the risk of pregnancy as a result of spontaneous resumption of ovarian activity.

Disclosure

The authors report no conflict of interest.

References

1. Rebar RW. Premature ovarian failure. Obstet Gynecol 2009; 113: 1355- 1363.

2. Nelson LM. Clinical practice. Primary ovarian insufficiency. N Engl J Med 2009; 360: 606-614.

3. Komorowska B. Autoimmune premature ovarian failure. Menopause Rev 2016; 15: 210-214.

4. Bidet M, Bachelot A, Bissauge E, et al. Resumption of ovarian function and pregnancies in 358 patients with premature ovarian failure. J Clin Endocrinol Metab 2011; 96: 3864-3872.

5. Tucker EJ, Grover SR, Bachelot A, et al. Premature ovarian insufficiency:

new perspectives on genetic cause and phenotypic spectrum. Endocr Rev 2016; 37: 609-635.

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