1 Department of Obstetrics and Gynecology, Turku University Central Hospital, Turku, Finland.
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Abstract |
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Key words: cervix/congenital cervical atresia/embryo transfer/IVF/pregnancy
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Introduction |
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In clinical practice, the aims of treatment of this condition are firstly to relieve the symptoms related to haematometra and retrograde menstruation, and secondly to restore fertility and regular bleedings. Reconstructive operations have been problematic due to severe complications, i.e. intra-abdominal infections, eventually resulting in hysterectomy in most cases (Maciulla et al., 1978, Rock et al., 1984
). Recently it has been suggested that a reconstructive operation may be worthwhile only in cases with cervical dysgenesis, but not with a complete absence of cervix (Jacobsen and DeCherney,1997).
We describe a successful pregnancy in a woman with cervical dysgenesis and hypofunctioning endometrium. The pregnancy was achieved after in-vitro fertilization (IVF) and an ultrasound-guided transmyometrial embryo transfer. To our knowledge this is the first reported case of a successful pregnancy after unsuccessful operative correction of congenital cervical atresia.
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Case report |
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The patient underwent two operations in order to build a functional uterovaginal passage. In January 1990, a colpoplasty was performed, but the attempt to create a canal through the cervical stoma was unsuccessful. Eight months later another attempt to reconstruct the uterovaginal canal was performed by laparotomy. Prior to the operation patient received oestrogen patches (100 µg/day) for 8 weeks in order to grow an endometrial lining and cause haematometra, making it easier to enter the uterine cavity. No endometrial thickening was observed, however. Operative exploration revealed a bicornuate uterus with a rudimentary left horn and a normal right Fallopian tube. Both ovaries were normal and no endometriosis was seen. A connection between the uterine cavity and the vagina was created by placing a rubber catheter through the fibrotic cervical tissue for 3 weeks. Unfortunately, the canal was stenosed 2 months later. Since no signs of haematometra or endometriosis were seen, it was concluded that the endometrium was hypofunctional, possibly due to an oestrogen receptor defect. The patient had not consented to hysterectomy because she felt well and had no menstrual problems.
Five years later in 1994, at the age of 30 years, the patient presented again at the reproductive endocrinology unit, because she wished to become pregnant and have a child. The serum FSH, thyroid-stimulating-hormone, and prolactin concentrations, as well as her partner's semen analysis were normal. The couple was informed of the risks of assisted reproductive techniques and the possible pregnancy. The couple refused the options of adoption and the use of a surrogate mother. A decision to attempt IVF and laparoscopic zygote intra-Fallopian transfer (ZIFT) was made. After pituitary down-regulation with nafarelin (Synarela®, Searle, Södertälje, Sweden), a total dose of 1125 IU of human menopausal gonadotrophin (Humegon®, Organon, Oss, The Netherlands) was used for ovarian stimulation. Ovum retrieval was performed by an ultrasound-guided vaginal puncture of follicles 36 h after i.m. administration of human chorionic gonadotrophin (HCG). Out of the four oocytes retrieved, three fertilized. At laparoscopy for ZIFT the right tube was observed to be occluded. The embryos were frozen at 2- to 4-cell stage, but failed to survive the thaw.
In September 1996, a second IVF cycle was performed using a mid-luteal down-regulation protocol with nafarelin (Synarela®, Searle). For ovarian stimulation 150 IU urinary FSH (Fertinorm HP®, Serono, Randolph, MA, USA) s.c. was used for 11 days. Oocyte retrieval was performed as described above. Five of eight oocytes were fertilized, and two 4-cell stage embryos were selected for ultrasound-guided transmyometrial embryo transfer, which was performed under general anaesthesia using a 6 MHz endovaginal ultrasound probe (Tosbee SSA-240-A, Toshiba, Tokyo, Japan) and a `Towako' embryo transfer catheter (Cook, Sydney, Australia) (Kato et al., 1993, Sharif et al., 1996
). For luteal support daily 600 mg micronized progesterone (Lugesteron®, Leiras, Turku, Finland) was administered vaginally. Fourteen days after the embryo transfer, serum HCG concentration was 588 U/l, and 2 weeks later, vaginal ultrasonography revealed an intrauterine gestational sac with fetal heartbeat.
The pregnancy progressed normally until 24 weeks gestation, when the patient was referred to the antenatal clinic for haematuria. A mild cystitis and bleeding inside the urethra were observed by cystoscopy. A possible invasion of placental tissue into the bladder was excluded by ultrasound, Doppler, and MRI examinations. An urinary infection was treated with pivmecillinamin 200 mgx3/day (Selexid®, Lövens, Ballerup, Denmark).
Three weeks later, the patient was admitted to the antenatal clinic for premature uterine contractions. The well-being of the fetus was monitored by repeated cardiotocography, ultrasound, and Doppler examinations. Since there was a high risk of premature delivery, maturation of fetal lungs was induced at 28 weeks gestation using weekly dexamethasone 8 mg i.m. injections (Dexametason®, Orion, Helsinki, Finland) for 5 weeks. The maturation of fetal lungs was confirmed by the measurement of lecitin/sphingomyelin ratio in amniotic fluid at 28 and 30 weeks gestation. At 31 weeks the patent exhibited symptoms of pre-eclampsia (headache and visual disturbances) with elevated blood pressure and oedema. Intravenous magnesium was administred, and because of rapidly progressing pre-eclampia a Caesarean section was performed a week later. A healthy boy weighing 1610 g was born in breech presentation (Apgar 6/7). The uterine cavity was carefully cleaned of decidua at the caesarean section. To reduce postpartum bleeding the patient received 100 mg medroxyprogesterone acetate i.m. (Depo-Provera®, Upjohn, Kalamazoo, USA). Both the mother and the baby recovered well and the postnatal phase was uneventful. One month after stopping breast feeding the patient was symptomless, and an ultrasound examination revealed a thin (3 mm) endometrium. One year later, the patient referred to the reproductive endocrinology unit, because of abdominal pain. A fluid-filled uterus, 7 cm in longitudinal diameter, was seen at ultrasound examination. The haematometra was successfully drained by a needle aspiration under ultrasound guidance and general anaesthesia. After the procedure, progestin treatment with lynestenol (Orgametril®, Organon) 10 mg daily was started to inhibit the proliferation of endometrium. The patient is currently considering a third IVF treatment.
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Discussion |
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After operative corrections of cervical atresia, four successful pregnancies have been reported previously (Table I) (Zarou et al., 1973
, Fraser, 1989
, Hampton et al., 1990
, Thjissen et al., 1990
). Two of them started spontaneously after reconstructive operations (Zarou et al., 1973
, Hampton et al., 1990
) and one after a prolonged treatment of endometriosis with danazol (Fraser, 1989
). A patient who became pregnant after a second IVF cycle with ZIFT has been described (Thjissen et al., 1990
). At the first unsuccessful IVF treatment the authors used transmyometrial embryo transfer, which they described as difficult due to a firm myometrium (Thjissen et al., 1990
). After the first report of successful transmyometrial embryo transfer using a spinal needle in humans (Parsons et al., 1987
), the equipment and technique for transmyometrial embryo transfer have been improved (Kato et al. 1993
). In patients with cervical stenosis, the `Towako method' has resulted in pregnancy rates comparable with those obtained by uncomplicated transcervical embryo transfers (Kato et al., 1993
, Sharif et al., 1996
).
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In patients with cervical atresia, the chance of a spontaneous pregnancy is reduced even after a successful reconstruction of the genital tract due to severe endometriosis, as well as cervical and tubal factors. However, the development of assisted reproductive techniques can now offer a real possibility of a pregnancy and a child of their own to these patients. Therefore, the patients should be carefully counselled when they present at the time of menarche, and hysterectomy should not be offered as the treatment of choice. In a case with normal endometrium, therapeutic amenorrhoea should be considered.
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Acknowledgments |
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Notes |
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3 To whom correspondence should be addressed at: Turku University Central Hospital, Department of Obstetrics and Gynecology, Kiinamyllynk. 48, FIN-20520 Turku, Finland
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References |
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Submitted on September 2, 1998; accepted on February 4, 1999.