The Use of the Transabdominal Cerclage in Women with Congenital Malformations of the Reproductive Tract

Case Report

The Use of the Transabdominal Cerclage in Women with Congenital Malformations of the Reproductive Tract

Corresponding author:Dr. Emma Mullen, Department of Obstetrics and Gynaecology, King’s College London, London. Tel: 07772151404; Email: e.e.mullen@icloud.com

Abstract

There are a vast spectra of reproductive tract malformations, many of which put women at increased risk of preterm birth. Improvements in identification and management of recurrent late miscarriages have been made and we propose that a regime of monitoring and appropriate intervention could benefit a much broader patient base, including those with congenital malformations. Many of these women have undergone surgical procedures to correct anatomical abnormalities; with the reconstructed tract alleviating the presenting symptoms but not necessarily providing a system compatible with term pregnancy.

A transabdominal cerclage aims to maintain a competent genital tract, until a time when a planned cesarean section of a term baby can be performed. We report the case of a 30-year old woman who underwent trans-vaginal correction of vaginal agenesis at 17-years old. She attended St Thomas’ Preterm Surveillance Clinic for insertion of a supportive transabdominal cerclage, and underwent subsequent in-vitro fertilization therapy. Following successful insemination and careful antenatal monitoring by the multidisciplinary team, she gave birth to a healthy term male.

Keywords: Transabdominal Cerclage; Vaginal Agenesis

Abbreviations: TAC – transabdominal cerclage; IVF – in-vitro fertilization; UTI – urinary tract infection

Introduction

The abdominal cerclage was originally introduced in 1965 by Benson and Durfree for use in patients in whom the cervix was too short to allow for a vaginal stitch [1]. This was either a consequence of previous surgical procedures or congenital anomalies. As this technique became popular and more widely practiced, indications broadened to include women who have had: wide or extensive cervical confiscation, cervico-vaginal fistulas, previous miscarriages, or a previously failed vaginal stitch [2]. As our understanding of the effects of reproductive tract abnormalities develops, coupled with improving surgical procedures and success rates, the indications for such supportive interventions continues to increase.

The transabdominal cerclage (TAC) aims to provide mechanical support to the cervix at the level of the internal so, to reduce the risk of pre-term delivery. This can be exploited in women with congenital malformations affecting the quality and competency of the reproductive tract to carry a fetus to term. It is thus hypothesized that this surgical technique may also improve the successful pregnancy rate in this population, with congenital anomalies. In the long term this may improve assisted pregnancy rates when used in high-risk patients alongside the developing surveillance techniques of monitoring cervical length and fetal fibronectin level.

The TAC procedure consists of a surgical suture or tape being passed around the cervico-isthmic junction, either via laparoscopy or laparotomy [2,3]. This can be done either pre-conceptually or in early pregnancy. A systematic review has found no statistical difference between these various practical details when regarding the endpoint of a successful term pregnancy to term [3]. In addition, across the methods, population-based cohort studies have reported success in carriage of up to 5 subsequent pregnancies [4]. Many of the preterm deliveries in these patients were associated with factors unrelated to the cerclage or cervical insufficiency.

Women with congenital malformations of the reproductive tract usually present one of two ways; early in adolescence with symptoms of cyclical pain and amenorrhea, or later with difficulties achieving and sustaining a pregnancy. For many of these women the main goal is to alleviate symptoms and ensure functionality of the reproductive tract at the crude level of pain-free intercourse and passage of menstrual blood [5]. Depending on the particular abnormality, the extent of repair and the method used varies [6]. The specifics of the malformation determine whether catheter dilatation is sufficient to open up a partially formed reproductive tract or whether neo-vaginal reconstruction must be used. The reconstruction can often lead to compromised functionality through new adhesions, muscle stiffness or direct trauma. These can affect any part of the tract, again posing issues in carrying a successful pregnancy. There may also be further problems to achieving pregnancy due to congenital issues with ovarian and uterine functionality.

Consequentially, for many of these women the suggested way to have a child is to use a gestational surrogate (if ovaries are functioning) or invasive IVF techniques involving trans-myometrium insertion of fertilized eggs [7,8]. The literature is limited regarding either of these strategies, with both relying on the preserved functionality of aspects of the reproductive tract, and posing their individual issues [9,10].

When considering the woman holistically, it is important to recognize the value in restoring functionality in early years, but also supporting a woman’s desire for pregnancy in later life [5]. Taking the patient from presentation to pregnancy is complicated, often requiring multiple medical and surgical interventions. The end goal is to reconstruct the reproductive tract to a state recognizable as “normal” and then to continue treatment until a successful term pregnancy is achieved.

Case Report

Our case is of a 30-year old female Nursery Nurse with an extensive history of gynaecological surgical interventions and reconstruction. She started menstruating at age 13 but never progressed to a regular or normal bleeding pattern. She originally presented to her GP with cyclical dysmenorrhea, irregular spotting and recurrent urinary symptoms.

At 14 years old the patient commenced the Microgynon combined oral contraceptive pill under the guidance of her GP. This was started in an attempt to alleviate the cyclical symptoms. She subsequently switched to Yasmin pill, which, unlike the preceding contraceptives, she felt gave her adequate symptomatic improvement.

Despite these simple and primary care-led interventions, the patient’s symptoms worsened with age. This led to frequent attendances to A&E, sometimes up to 2-3 times per month. With each admission, she was treated with antibiotics for symptomatic bacteriuria although this did not give her even temporary symptom resolution.

At this point, the differential diagnoses of appendicitis, recurrent UTI, bicorn ate uterus, endometriosis, ovarian cysts and torsion were all considered. The patient herself had no past medical history of note and a family history only significant of menorrhagia and recurrent UTIs in first-degree female relatives. Working with this background, examinations, blood tests, and scans were performed. These proved inconclusive and the patient consequently underwent a diagnostic laparoscopy, during which obstructed fallopian tubes were identified and treated.

In spite of correction, symptoms returned and began to worsen. Further MRI scans and surgical investigations diagnosed a congenital malformation of the reproductive tract. Although the clinical picture was not entirely clear, the impression was that the patient had vaginal agenesis. By this time the patient had already undergone 4 diagnostic laparoscopies with consequential removal of multiple cysts and unblocking of her fallopian tubes.

At 17-years old the patient was treated at UCLH with a trans-vaginal procedure to correct a low, partial agenesis of the vagina. Despite this hypoplasia of the vagina, an apparently normal ant everted uterus was identified, through an incomplete cervix.

Even after this drastic surgical intervention, the patient experienced mild cyclical pain and irregular bleeding. In 2010, it was found that the artificially created cervix had tightened and thus led to increased pressure within the uterus with each menstruation. Cervical dilatation was trialed to relieve the pain and regulate bleeding. A continuing symptom profile and recurrent haemosalpinges that necessitated laparoscopic intervention, meant that in 2014 the patient underwent an elective bilateral salpingectomy.

In 2015, the patient’s menstrual cycles were 32 days long with 7 days of bleeding. At this point she was only experiencing mild pain with each bleed. She began taking folic acid supplements with the intent of assisted conception in the future.

The patient was referred to Guy’s and St. Thomas’ for fertility treatment. The patient’s uterus and ovaries were recognised to be functional normal. Due to the lack of fallopian tubes and the re-constructed nature of the vagina and cervix, pregnancy without the use of further interventions would not be possible. The artificial nature and, therefore, inherent incompetence of the cervix would likely prove inadequate to support a pregnancy to term. As a result, a pre-emptive abdominal cerclage procedure was suggested and discussed with the patient.

The patient was scheduled to commence the first cycle of IVF in the second week of January 2017, given a full recovery from the successful laparotomy and TAC. Her partner’s sperm had previously been analysed to ensure that this would not cause further unforeseen issues in establishing pregnancy.

In December 2016 the patient had a TAC placed in-situ. The procedure was undertaken by Professor Andrew Shennan at St. Thomas’ Hospital with the assistance of his research fellows and clinical team. As the patient is only 30 years old, both the legislation surrounding IVF in her area of residence, and the increased risk of preterm delivery with twin pregnancies, meant that only one embryo was inserted into her uterus. This was successfully transferred and the other embryos were frozen with the intent of future cycles if this one were to be unsuccessful, or the patient wished to pursue further pregnancies.

The patient’s pregnancy progressed well and she was regularly followed-up to ensure growth of the fetus was within normal parameters and that the modified reproductive tract was adapting well to the pregnancy. Her cervical length and quantitative metal fibronectin were monitored at regular intervals between 18 and 24 weeks’, and found to be stable and reassuring. The extensive surgical interventions this patient has had over the preceding 10 years, as well as the abdominal cerclage necessitated a planned caesarian section was agreed between the patient, her partner and the multidisciplinary team at 38 to 39 weeks’ gestation. A healthy male infant was delivered and both the patient and her child are doing well.

Conclusion

Errors in the complex fetal development of the paramesonephric ducts and urogenital sinus lead to a broad spectrum of congenital abnormalities. Any of the anatomical entities of the reproductive tract can fail to appropriately develop, due to incomplete fusion of these primitive ducts. The vast spectrum of reproductive tract malformations can lead to women who are often unable to carry a successful pregnancy to term. In light of the complexities and subtleties of the anatomy it can be hard to identify and therefore treat the exact underlying pathology. Many of these women undergo procedures to correct abnormalities. The reconstructed tract aims to alleviate the presenting complaint; allowing normal intercourse and menstruation while preserving fertility [5,6]. Despite this, it does not guarantee a system compatible with carrying a pregnancy to term [9,10]. This inadequacy is, in part, often due to the incompetence of the constructed cervix to maintain a sterile and closed uterine cavity.

A TAC aims to maintain a competent cervix artificially, until a time when a planned caesarean section can be performed. The trans-abdominal approach allows a more thorough assessment of the health of the reconstructed tract and for high placement of the stitch, at the level of the internal so. Both of these aim to improve outcomes and guide future interventions [3,4].

There has been considerable improvement in the success of reconstructive surgery. This is in part due to more accurate identification and diagnosis of abnormalities, particularly the utilization of high resolution scanners. Secondly, this can be attributed to the advancement in surgical techniques, equipment and recovery [3,10]. As many of these women have further fertility issues, IVF is often indicated, at great emotional and financial costs. The use of the TAC may prove to support these women to a natural term pregnancy while improving the long term success of fertility interventions.

This case study exemplifies how the indications in which we utilize pregnancy sustaining surgical strategies could be considerably broader. Vaginal agenesis is a particularly rare reproductive tract malformation, affecting only 1 in every 4000-10000 live born females, with only 7-8% possessing a functional uterus9. It is therefore apparent that vaginal agenesis in combination with a functioning uterus is rarely encountered clinically. Despite our patient’s rare presentation and combination of abnormalities, preterm delivery is unfortunately common, and the success of this treatment in her particularly complex case confirms how simple surveillance and reactive treatment if needed could benefit a larger group of women. It also confirms that IVF rates are unaffected by the surgical cerclage.

Exploiting a pregnancy-sustaining strategy may prove beneficial both emotionally and financially when considering multiple failed attempts at giving birth, including those involving invasive fertility management. Taking a pre-emptive approach, and providing regular surveillance and support as in our case, may alleviate subsequent emotional and medical morbidity.

References

  1. Benson RC, Durfee RB. Transabdominal cervicouterine cerclage during pregnancy for treatment of cervical incompetency. Obstetrice and Gynaecology.  (1965), 25: 145-155.
  2. Novy MJ. Managing reproductive failure by transabdominal isthmic cerclage. Contemp Ob/Gyn. (1977) 10: 17.
  3. Jazayeri A. Cervical Cerclage [online]. Medscape 2017.
  4. Lotgering F, Gaugler-Senden I, Lotgering S, Wallenburg H. Outcome After Transabdominal Cervicoisthmic Cerclage. Obstetrics and Gynaecology. (2006), 107(4): 779-784.
  5. Edmonds K. Congenital malformations of the genital tract and their management. Best Practice & Research Clinical Obstetrics & Gynaecology. (2003), 17(1): 19-40.
  6. Bates W, Wiser W. A technique for uterine conservation in adolescents with vaginal agenesis and a functional uterus. Obstetrics and Gynaecology. (1985), 66(2): 290-294.
  7. Wood E, Batzer F, Corson S. Ovarian response to gonadotrophins, optimal method for oocyte retrieval and pregnancy outcome in patients with vaginal agenesis. Human Reproduction. (1999), 14(5): 1178-1181.
  8. Esfandiari N, Claessens EA, O’Brien A, Gotlieb L, Casper RF. Gestational carrier is an optimal method for pregnancy in patients with vaginal agenesis (Rokitansky syndrome). International Journal of Fertility and Women’s Medicine. (2004), 49(2): 79-82.
  9. Moura M, Navarro P, Nogueira A. Pregnancy and term delivery after neovaginoplasty in a patient with vaginal agenesis. International Journal of Gynecology & Obstetrics. (2000), 71(3): 215-216.
  10. Liu Y, Wang F-Y. Successful vaginal delivery at term after vaginal reconstruction with labium minus flaps in a patient with vaginal atresia: A rare case report. The Journal of Obstetrics and Gynaecology Research. (2017), 43(7): 1217-1221.

Be the first to comment on "The Use of the Transabdominal Cerclage in Women with Congenital Malformations of the Reproductive Tract"

Leave a comment

Your email address will not be published.


*