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Intraligamentary extrauterine pregnancy delivered at term: a case report and review of literature.

Introduction

The diagnosis of intraligamentary extrauterine pregnancy is an enigma to the diagnostic acumen of the clinicians. The problem is further compounded in the environment such as ours where majority of pregnant women do not seek antenatal care and only present to the hospitals when complications have set in. Intraligamentary pregnancy is rare, it occurs once in approximately 245 ectopic pregnancies (1).

We report a case of intraligamentary pregnancy delivered at 41 weeks gestation in Nigeria with good maternal outcome and fully formed and grossly normal term fresh stillborn male baby.

Case Report

Mrs. S.A. was a 33 year old Gravida 4 para 2 with 2 children alive. She was referred from a Basic Health Centre (BHC) to Ekiti State University Teaching Hospital in Ado-Ekiti, Ekiti State, Southwest Nigeria, on the 3rd of August, 2013 at 16.00hrs. She complained of abdominal pain and ten hours history of inability to perceive fetal movement. She had regular menstrual cycles of 4 days in a 28days cycle prior to conception. The first day of her last menstrual period was 18th of October 2012 and the estimated gestational age at presentation was 41 weeks and 2 days. She had registered for antenatal care at the referring BHC at estimated gestational age of 36weeks. Her booking parameters as stated in the referral note were within normal limits. She had her first ultrasound scanning at 37 weeks, which reported a viable intrauterine pregnancy with estimated gestational age compatible with 37 weeks gestation and breech presentation. Her antenatal period had been without complications till the time of referral.

She had carried two pregnancies to term with one spontaneous abortion. Her first delivery was a live male child in 2009 by emergency caesarean section in 2009 due to intrapartum fetal distress. In 2011, she had a successful Vaginal Birth after Caesarean Section of a live female child. She was clinically stable on examination. She was not pale and her vital signs were within normal limits. The cardiovascular and respiratory systems did not reveal any abnormality. Abdominal examination reveals a healed Pfannensteil scar, symphysio fundal height of 38cm (which was compatible with term gestation), longitudinal lie, breech presenting fetus. There were no palpable uterine contractions. Fetal heart sound was absent. She had normal vulva, a posteriorly located closed cervix about 2cm long. There was no vaginal bleeding. She had a repeat ultrasound scanning at presentation which reported an intrauterine gestation at term with breech presentation, severe oligohydramnios and no fetal cardiac activity.

We made a diagnosis of a term intrauterine fetal death with a previous caesarean section scar. Packed cell volume was 39%; blood group was O Rhesus positive, all other results were essentially normal. An attempt was made to ripen her cervix with transcervical extra-amniotic Foley's catheter followed with oxytocin induction of labour which was unsuccessful. She was then planned for abdominal delivery. Anaesthesia was induced with sodium thiopental followed by succinylcholine for endotracheal intubation. Maintenance anesthesia was with nitrous oxide and oxygen. Intraoperatively, we found an intact uterus about 16 week's size on the infero-lateral side of the gestation.

The fetus was found within the two leaves of the right broad ligament, [Figures 1-2] surrounded by intact amniotic membranes.

The placenta was attached to the posterior aspect of the right broad ligament and the floor of the cavity in the broad ligament. The right ovary was compressed on the right pelvic side wall; the right tube was not distinctly visualized. The anterior leave of the broad ligament was incised and a fresh male still-born weighing 2.6kg was delivered [Figure 3]. The right adnexum was removed with the placenta. [Figure 4] The left ovary and tube were grossly normal. Hemostasis was secured and a rubber drain left in-situ. Blood loss was estimated at 2500ml. She had 3 units of blood transfused. Her recovery was satisfactory; post operative packed cell volume was 30%. She was discharged from the ward on the fourth post operative day.

[FIGURE 1 OMITTED]

A written informed consent was given by the patient to publish these records and pictures. Ethical approval was also obtained from the institution's ethics and research committee.

Discussion

Intraligamentary ectopic pregnancy is a retroperitoneal abdominal pregnancy in which the product of conception grows within the two leaves of the broad ligament. It is a rare pregnancy with an incidence of about 1 in 183,900 pregnancies (2). This is to the best of our ability the first report of this condition from our centre, and there have been very few reports from other centres in Nigeria (3,4); which underscores the rarity of intraligamentary pregnancy. Two theories had been proposed to explain the pathogenesis of Intraligamentary pregnancy otherwise referred to as broad ligament ectopic pregnancy. Kobak et al (5) explained that the majority of intraligamentary pregnancies occur as a result of the rupture of tubal ectopic pregnancies along the mesosalpingeal border with secondary escape and implantation of the fertilized ovum between the two leaves of the broad ligament. Paterson and Grant (6) having noted cases with normal Fallopian tubes believed that intraligamentary pregnancy occurs from primary implantation of the fertilized ovum on the broad ligament with burrowing between its two leaves. Intraligamentary pregnancy can also occur if a uterine fistula develops between the endometrial cavity and retroperitoneal space (7). The first theory probably explains the pathogenesis of the broad ligament pregnancy in our patient because the Fallopian tube on that side was not demonstrated intact. Though she had a previous caesarean section, there was no evidence of fistulous opening between the endometrial cavity and the retroperitoneal space.

[FIGURE 2 OMITTED]

[FIGURE 3 OMITTED]

[FIGURE 4 OMITTED]

The diagnosis of intraligamentary pregnancy is usually made in advanced pregnancy as occurred in our patient. This is more likely to be so in Nigeria where more than 40% of pregnant women do not seek antenatal care but only report when there is occurrence of complications (3,4,8) as was seen in this case. Mrs. SA was only referred to our facility after failure of onset of spontaneous labour at 41 weeks gestation, for possible delivery. Early diagnosis is essential because catastrophic complications can occur due to early separation of the placenta. The associated maternal and perinatal mortality are 40% and 95% respectively (9,10). There was no loss of maternal life in this case, there was however perinatal death. Ultrasonography remains the main method for the diagnosis of extra uterine pregnancy (8), however it can be disappointing in advanced pregnancy as occurred in this case.

Other radiological studies such as MRI and CT scan are helpful in the diagnosis (11) but are not usually available in most part of the developing world (3,4,8). In poor resource settings, a high index of suspicion is essential for prompt diagnosis and timely intervention to prevent catastrophic consequences. The findings of abdominal pain, abnormal lie and presentation, absent fetal heart tone, closed uneffaced cervix and the non responsiveness of the uterus to intravenous oxytocin heightened our suspicions of this condition. The diagnosis was confirmed intraoperative with the finding of a fresh grossly normal stillborn confined between the two leaves of the broad ligament and containing the placental. This conforms to the history of ten hours history of last perception of fetal movement and ultrasound scanning findings of IUFD. Bleeding from the placenta implantation site is the most life threatening complication encountered at laparotomy and the decision to remove the placenta or not can be a determining factor for survival of the woman. It is recommended to leave the placenta in situ if removal is life threatening and follow up the patient with post-operative Methotrexate therapy, hCG estimation and ultrasonography (12). Chemoembolization through lateral sacral artery has also been advocated (13). In this case however, there was significant bleeding from the detached placenta portions that necessitated the removal of these portions to secure haemostasis.

Conclusion

We present a case of an advanced intraligamentary pregnancy that would have led to the delivery of a healthy baby boy if the patient had presented promptly. The diagnosis of intraligamentary pregnancy requires a high index of suspicion; therefore medical practitioners should consider it a possible differential diagnosis. Ultrasonography and Magnetic resonance imaging are diagnostic tools that may help the physician in preoperative diagnosis. We also advocate early pregnancy diagnosis and community education on the importance of antenatal care in the developing countries to reduce the mortality and morbidity from ectopic pregnancy.

Conflict Of Interest

The authors declare no conflict of interest.

Contribution of Authors

Ade-Ojo, Akintayo and Aduloju all contributed to the surgery. Afolayan was the anaesthetist at the surgery. Akintayo and Ade-Ojo were the major contributors to the manuscript, Olagbuji assisted in literature search. All authors read and approved the final manuscript.

References

(1.) Ziek HK, Miyazaki FS, Baker HT, White JD. Advance Interligamentary Pregnancy: Report of a case with survey to date. Obstet Gunyaecol, 31(5); 1968: 643-648.

(2.) Cordero DR, Adra A, Yasin S, O'Sullivan MJ. Intraligamentary pregnancy. Obstet Gynaecol Survey 1994; 49(3): 206-9.

(3.) Abdul MA, Tabari AM, Kabiru D, Hamidu N. Broad Ligament Pregnancy: A Report of Two Cases. Ann Afri Med, 7 (2) 2008: 86-87.

(4.) Ikechebelu JI, Onwusulu DN, Chukwugbo CN. Term Abdominal pregnancy misdiagnosed as Abruptio Placenta. Nig J Clin Pract 2005; 8(1): 43-45.

(5.) Kobak AJ, Fields C, Pollack SL. Intraligamentary Pregnancy: The extraperitoneal type of abdominal pregnancy. Am J Obstet Gynecol. 1955; 70-175.

(6.) Paterson WG, Grant KA. Advanced intraligamentous pregnancy; Report of a case, review of the literature, and a discussion of the biological implications. Obstet Gynecol Surv. 1975; 30: 715.

(7.) Rudra S, Gupta S, Taneja BK, Garg M. Full-term broad ligament pregnancy. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010329.

(8.) Nnadi D, Nwobodo E, Ekele B. Abdominal pregnancy in Usmanu Dan-Fodiyo university teaching hospital, Sokoto: A 10-year review. J Basic Reprod Clin Sci 2012; 1: 34-7.

(9.) Dahab AA, Aburass R, Shawkat W, Babgi R, Essa O, Mujallid RH. Full-term extrauterine abdominal pregnancy: a case report. J Med Case Rep 2011; 5:531.

(10.) Onan MA, Turp AB, Saltik A, Akyurek N, Taskiran C, Himmetoglu O. Primary omental pregnancy: case report. Hum Reprod 2005; 20:807-9.

(11.) Sharma S, Pathak N, Goraya SPS, Mohan P. Broad ligament ectopic pregnancy. Sri Lanka J Obstet Gynaecol 2011; 33: 60-62

(12.) Gupta P, Sehgal A, Huria A, Mehra R. Secondary abdominal pregnancy and its associated diagnostic and operative dilemma: three case reports. J Med Case Reports 2009, 3:7382 doi: 10.4076/1752 1947-3-7382

(13.) Zhang J, Guan S. Chemoembolization through lateral sacral artery to treat uterus broad ligament pregnancy. Life Science Journal. 2012; 9(2):36-38

Idowu P Ade-Ojo [1], Akinyemi A Akintayo [1] *, Jide M Afolayan [2], Olusola P Aduloju [1] and Biodun N Olagbuji [1]

Department of Obstetrics and Gynaecology, Ekiti State University, Ado-Ekiti [1] ; Department of Anaesthesia, Ekiti State University, Ado-Ekiti [2].

* For Correspondence: Email: akinyemiakintayo@yahoo.co.uk, akinyemiakintee@gmail.com; Phone: +234803-376-7542.
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Article Details
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Title Annotation:CASE REPORT
Author:Ade-Ojo, Idowu P.; Akintayo, Akinyemi A.; Afolayan, Jide M.; Adulojuand, Olusola P.; Olagbuji, Biodu
Publication:African Journal of Reproductive Health
Article Type:Report
Date:Mar 1, 2016
Words:1824
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