Simultaneous pregnancy in each uterine cavity of a double uterus in a young Nigerian multipara who presented with a retained second twin following an unsupervised preterm labor at home; Case report.

INTRODUCTION: Simultaneous pregnancy in each uterine cavity of a double uterus is unusual but is a recognized risk factor for preterm labour and other poor obstetrics outcomes. The work has been reported in line with the SCARE criteria. PRESENTATION OF CASE: We report an unusual case of simultaneous pregnancy in each uterine cavity of a double uterus in a young African grand multipara who presented with a retained second twin following a preterm labour at home. DISCUSSION: A double or didelphys uterus as reported in the literatures is still uncommon even in Africa. While infections are very important and always considered causes of preterm labour a high index of suspicion will help give a diagnosis of a uterine anomaly and this will lead to more precise clinical examinations and studies in cases of recurrent miscarriages and preterm birth where other causes such as infection and cervical incompetence has been ruled out.
CONCLUSION: Double uterus is an important cause of recurrent preterm labourv and miscarriages as seen in the index case. Thorough pelvic examination should be conducted for women of reproductive age groups when they present for gynecological consultation to rule out the rare occurrence of double uterus and other uterine abnormalities. Health education should be intensified through different media on the reality of double uterus and its attendant complications as a means to boost ante natal care booking and attendance for early diagnosis and appropriate management of this congenital anomaly.

Introduction

Simultaneous pregnancy in each uterine cavity of a double uterus is unusual but was reported by Davies and Cellan-Jones in 1927 [1] and recently by Yang et al. in 2015 [2]. A failure in Mullerian duct fusion might result in a didelphys uterus, as well as a complete or partial septate vagina [3]. Its prevalence is reportedly higher among infertile women compared to the general population [[2], [4]]. Congenital anomalies of the uterus including double uterus is a recognized risk factor for preterm labour and other poor obstetrics outcomes [[5], [6]]. We report an unusual case of simultaneous pregnancy in each uterine cavity of a double uterus (Fig. 1) in a young African grand multipara who presented with a retained second twin following a preterm labour at home. The work has been reported in line with the SCARE criteria [7].

Fig. 1

A double or didelphic uterus.

Presentation of case

We present the case of an un-booked Para 3+8, 1alive who was brought to the maternity section of a Hospital at seven and half month gestation with 12 h history of retained second twin. The first twin was delivered following unsupervised labour home. She had recurrent second trimester miscarriages and preterm births at home due to undiagnosed causes. She has not had any clinical or ultrasound diagnosis of an abnormal uterus. She never attended antenatal care and was not on any regular medications. There was no history of hypertensive, diabetic, twinning and congenital anomalies. She was said to have been bleeding after delivery of the first twin. The first twin appeared premature and weighed 1.2 kg. It was a female baby. There was no history of instrumentation or ingestion of any traditional medication. She was said to be tired and made scanty urine. There was no other significant family or drug history.

Examination findings showed a young woman who was in labour pains. She was warm to touch, pale and dehydrated. There was no pedal edema. The abdomen was gravid and symphysis-fundal height was 28 cm. The fetal heart tone was absent. The vulva and vagina were normal. The umbilical cord hanging out from the vagina was unclamped and one cervical os was felt fully. Another cervical os was barely felt but was closed. A diagnosis of retained dead second twin and maternal distress was made.

She was actively resuscitated with intravenous fluid, blood and had antibiotics. Consent was obtained for an emergency caesarean section under general anesthesia. Operative findings showed clean peritoneal cavity and double uterus. Each uterus have a fallopian tube and ovary on its side (Fig. 2). The uteruses are not joined to one other (Figs. 3 & 4 ). The left uterus from which spontaneous vaginal delivery was effect was undergoing involution but still contained the placenta as evidenced by the umbilical cord protruding through the vaginal (Figs. 3 & 4). A female fresh still born weighing 1.1 kg was delivered from the right uterus. There were two separate placenta in each uterus. Estimated blood loss was 700Ml. There was no renal anomaly. Post-operatively she did well and was discharged after 5 days and advised for gynecological consultation regarding her condition. She was satisfied with her experience of care.

Fig. 2

Intraoperative Images Showing the fallopian tube of the left sided uterus following entrance into the abdomen, each uteri had only one fallopian tube.

Fig. 3

Intraoperative Images Showing highly vascular but poorly formed lower uterine segment of the right sided uterus before uterine incision.

Fig. 4

Intraoperative Images Showing the right-sided uterus after uterine incision and delivery of the retained twin.

Discussion

A double or didelphys uterus as reported in the literatures is still uncommon even in Africa. It will even be more unpopular because of poor care-seeking behavior and lack of diagnostic equipment. The large rural population, poverty and different phases of obstetrics delay also means that women needing care do not get access to quality care. While infections are very important and always considered causes of preterm labour a high index of suspicion will help give a diagnosis of a uterine anomaly and this will lead to more precise clinical examinations and studies in cases of recurrent miscarriages and preterm birth where other causes such as infection and cervical incompetence has been ruled out. This is in consonance to previous reports [[8], [9]]. In this case it assumed a clinical importance because of its predisposition to recurrent preterm labour and retained death second twin. Surprisingly each uterus contain fetuses of same sex and assumingly of same gestational age. Previous studies had reported superfetation of interval separated twins following spontaneous and assisted reproduction [[10], [11]]. Although chorionicity was not determined the authors have assumed that the fetuses were dichorionic. It was important to rule out renal anomalies because a didelphys uterus has been shown to be associated with a very rare congenital anomaly of the urogenital tract known as Herlyn-Werner-Wunderlich (HWW) syndrome, with ipsilateral renal anomaly [12].

Conclusion

Double uterus is an important cause of recurrent preterm births and miscarriages. Thorough pelvic examination should be conducted for women of reproductive age groups when they present for gynecological consultation to rule out double uterus. In the absence of this, pregnant women should have at least one ultrasound study to check their babies and their uterus for rare conditions in order to avoid the obstetrics catastrophe which was reported in our practice. Most importantly, health education should be intensified through different media on the reality of double uterus and its attendant complications as a means to boost ante natal care booking and attendance for early diagnosis and appropriate management of this congenital anomaly. Treatment should be individualized and outcomes and outcomes explored.

Conflicts of interest

The authors have no conflicts of interest to declare.

Funding

The authors have received no funding for their research.

Ethical approval

Ethical approval was obtained from Human Research Ethics Committee of the Kano State Ministry of Health with reference number: MOH/Off/797/T.1/195 and dated 29th September, 2017.

Consent

The patient has unconditionally given informed consent to the authors to report the findings. There has not been any forms of alterations.

Author contribution

Study concept or design: Emmanuel Ugwa and Emmanuel Ani.

Data collection, data analysis or interpretation: Emmanuel Ani, Iwasam Agbor, Emmanuel Ugwa.

Writing the paper: Emmanuel Ani, Iwasam Agbor, Emmanuel Ugwa.

Registration of research studies

This study has not been registered.

Guarantor

Emmanuel Ani