Abstract
A 34-year-old woman was referred to the in vitro fertilisation (IVF) clinic following failure to conceive after ovulation induction with antioestrogens and intrauterine insemination. She had a long history of hypothalamic amenorrhoea secondary to weight loss and stress and received hormone replacement to maintain her bone density. She also underwent radical trachelectomy and bilateral laparoscopic pelvic node dissection as fertility sparing surgery for cancer of the cervix stage 1B. She remained under our care for 4 years during which she had two successful IVF pregnancies with elective single embryo transfers on both occasions. She delivered preterm by caesarean section at 31 weeks and 35 weeks, respectively, for premature rupture of membranes with good outcomes. There was no evidence of local or distant recurrence of her early cervical cancer at 10-year follow-up at the combined gynaecology oncology clinic and she was discharged to primary care for follow-up.
BACKGROUND
A 34-year-old woman, para 0+1 with long history of secondary amenorrhoea and previous radical trachelectomy and abdominal cervico isthmic cerclage, attended the subfertility clinic in 1999.
CASE PRESENTATION
Presenting features
The patient had had 12 cycles of ovulation induction with antioestrogens (clomifene and tamoxifen) and in addition also had had 4 cycles of intrauterine insemination. In spite of confirmed ovulatory cycles she failed to conceive and was referred for in vitro fertilisation (IVF) in 2001. Assisted conception with elective single embryo transfer was undertaken to reduce the risk of complications associated with multiple pregnancy.
Past medical history
She had her menarche at the age of 12. At age 21, she lost 9 kg in weight and developed secondary amenorrhoea. Investigations revealed that she had hypogonadotropic hypogonadism secondary to weight loss (body mass index (BMI) 18) and stress. A diagnosis of squamous cell carcinoma of cervix stage 1 B was made in 1998 from a routine cervical screening. As the patient was very keen to have fertility preservation surgery, she was referred to a French clinic for a radical trachelectomy and bilateral laparoscopic pelvic lymphadenectomy instead of radical hysterectomy for her early stage cervical cancer.
A transabdominal cervico isthmic suture was also undertaken as a permanent cerclage. She was started on hormone replacement therapy to maintain her bone density and followed up regularly at the combined gynaecology oncology clinic.
INVESTIGATIONS
A dual energy x ray absorptiometry (DEXA) scan was carried out to check for bone mineral density in 1997 and this showed osteopoenia in her hip and spine. She was started on hormone replacement therapy. She had further follow-up DEXA scans in 2004 and 2007, which confirmed osteopoenia. The couple had fertility investigations and these were normal.
TREATMENT
The patient underwent a total of five cycles of fresh stimulated cycles of in vitro fertilisation (IVF) and three cycles of frozen embryo thaw cycles between January 2002 and July 2006. Adequate priming of endometrium was undertaken with 2 months of cyclical hormone replacement therapy before starting IVF treatment. She had an elective single embryo transfer with each cycle and managed to conceive twice with stimulated IVF cycles. Both her pregnancies were treated as high-risk pregnancies and were monitored closely for preterm labour. She had uncomplicated first and second trimesters in both pregnancies. Because of premature rupture of membranes in both her pregnancies, she delivered by caesarean sections at 31 weeks (2004) and 35 weeks gestation (2007), respectively, with good outcomes.
OUTCOME AND FOLLOW-UP
At her recent follow-up in 2008, she was entirely well and asymptomatic. There was no evidence of local or distant recurrence of the early cervical cancer. She was discharged from the gynaecology oncology clinic and will return on routine recall for the cervical screening programme until the age of 60. She does not wish to go through further IVF but would be delighted if she were to conceive naturally. She plans to continue her hormone replacement therapy for treatment of her osteopoenia.
DISCUSSION
This may be the first reported case of a woman with hypothalamic amenorrhoea and radical trachelectomy conceiving from IVF treatment. The first successful radical vaginal trachelectomy for invasive cervical carcinoma was described and published by Dargent in 1994. The operation included a laparoscopic pelvic lymphadenectomy, which was followed by the removal of cervix together with surrounding parametria in order to protect corpus of the uterus and the ovaries.1 Radical trachelectomy is an operation developed as an alternative to radical hysterectomy for patients with small volume, early stage cervical cancer who wish to retain their fertility. Patients should be preoperatively informed about the risks of infertility, early pregnancy loss, preterm delivery and neonatal complications.1 Patients who have undergone radical trachelectomy may rarely request assisted conception. Kay et al reported a successful pregnancy following radical trachelectomy with egg donation and IVF.2 Jolley et al, in their review of literature revealed that 40% of women conceived following radical trachelectomy. They reported first and second trimester pregnancy loss of 33%, preterm delivery rate of 25% and 42% culminated in delivery of a live born infant at term. They also noted that the majority of successful pregnancy outcomes have occurred with a cerclage in place.3 Plante et al reported a twin pregnancy following IVF and this woman elected to have an embryo reduction and subsequently delivered at term.4 Ramirez et al analysed the published literature and concluded that radical trachelectomy is safe and feasible and pregnancy outcomes are very favourable. In this review of 520 patients, 43% of patients who underwent a radical trachelectomy subsequently attempted to become pregnant and 70% of these women were successful at achieving a pregnancy. They recommended that all patients wishing to preserve fertility who have a diagnosis of cervical cancer should be encouraged to discuss radical trachelectomy with their gynaecologist.5
LEARNING POINTS
Radical trachelectomy should be offered to selected patients with early stage cancer of the cervix.
Elective single embryo transfer should be offered in women with radical trachelectomy to reduce the complications in pregnancy.
Priming of the endometrium with cyclical hormone replacement therapy should be offered to women with hypothalamic amenorrhoea prior to in vitro fertilisation (IVF) treatment to increase their chance of pregnancy.
Women seeking fertility investigations with history of radical trachelectomy should be referred to a specialist infertility clinic for management.
Acknowledgments
We thank all the staff in assisted conception programme for their care of this patient during her treatment.
Footnotes
Competing interests: None.
Patient consent: Patient/guardian consent was obtained for publication.
REFERENCES
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