Broad Ligament Pregnancy: Facing the Battle Unarmed.

Here we discuss a case of severe oligoamnios, its course, and the final unpredictable outcome. A 30 years old lady was admitted to our casualty with the diagnosis of gravida 2, para 1, live birth 1 with 24 weeks of gestation with low lying placenta with anhydramnios. Her first visit was at 23 weeks at a government hospital from where she was referred to our hospital in view of anhydramnios. She had no history of leaking, bleeding per vaginum, fever, pain in abdomen. Past medical history was not significant. Obstetric history: she had a full-term normal vaginal delivery of healthy male baby 13 years ago. Her present pregnancy was a spontaneous conception. She had no history of dilatation and curettage in the past. There was no history of use of any intrauterine contraceptive device or oral/injectable contraceptive. On admission, her vitals were stable, she was afebrile, pulse was 90 beats per minute (bpm), blood pressure was 110/70 mmHg. She had no pallor, icterus, or pedal edema. Systemic examination was normal. The results of abdomen examination: fundal height was 24 weeks, uterus was non tender, non tense and relaxed, liquor was reduced grossly, fetal parts were palpable, fetal heart sound was present and regular on auscultation. The results of speculum examination: no leaking or bleeding was observed. Laboratory investigations showed hemoglobin was 116 g/L, total leucocyte count was 13.2 × 10 9 /L, neutrophils were 78%, lymphocytes were 14%, and platelet count was 190 × 10 9 /L. Obstetric ultrasound revealed a single live intrauterine fetus of 23 weeks of gestation with absent amniotic fluid, placenta anterior, partially covering cervical os, no gross fetal anomalies. High vaginal swab and urine culture was sterile. Relatives and patient were counseled about the risk of extreme prematurity and placenta previa. She was planned for conservative management and steroid cover was given. Maternal and fetal monitoring was done with alternate day amniotic fluid index, biweekly complete blood count, C reactive protein, and weekly high vaginal swab for culture. Conservative management was continued for 4 weeks. After 4 weeks of conservative management, at 28 weeks of gestation, patient complained of pain in the lower abdomen. She had 2 spikes of fever (38.3 °C), not associated with chills and rigors. General condition of the patient was fair, pulse was 120bpm, blood pressure was 110/68 mmHg, no pallor. Abdomen examination showed uterus of fundal height 26–28 weeks, relaxed but uterine tenderness was present, fetal heart rate was 180 bpm, regular. Local examination still showed no bleeding or leaking. All routine investigations were sent. Hemoglobin was 106 g/L, total leucocyte count was 21.5 × 10 9 /L, neutrophils were 90%, lymphocytes were 6%, platelets were 230 × 10 9 /L, C reactive protein was positive. In view of this clinical presentation, a diagnosis of possible chorioamnionitis was made and the patient was shifted for cesarean in view of type II placenta previa and chorioamnionitis after explaining maternal and fetal risks and counseling the patient and relatives about the materno-fetal outcome possibilities. Peroperatively, a thick-walled sac was seen. Omentum and bowel were adhered to the lateral and postero-superior surface of the sac preventing delineation of its boundaries. The presentation raised high suspicion of uterine anomaly/secondary abdominal pregnancy. In this anticipation, taking precautions, the anterior surface of the sac was incised. A live female baby of 760 g was delivered. Placenta could be seen lying on left lateral wall of the sac. The normal uterine musculature and wall could not be defined. This led to the exploration of the abdomen. Sigmoid colon and terminal ileum were adherent to the sac. Adhesiolysis was done. On insinuating hand posterior to the sac, a firm muscular structure was felt posteriorly towards left pelvic wall which was brought towards incision site and identified as uterus which was bulky. The gestational sac on the right side was identified in between the leaves of the broad ligament and diagnosed as an intraligamentary pregnancy. Placenta was partially separated and placental bed was bleeding. Placenta was extracted and hemostatic sutures were taken in the bed. Right fallopian tube was identified on the upper aspect of the sac. Right ovary was adherent to the sac and was dissected and salvaged. The uterus left-sided fallopian tube and ovary were normal. Sac was excised and margins secured after extraction of the baby and placenta (Fig. 1 ). Multiple hemostatic sutures were applied to achieve hemostasis of the placental bed. The wall of the sac was excised with sequential clamping, cutting, and suturing of the margins avoiding base of the broad ligament (Fig. 2 ). Duration of surgery lasted two and a half hours and patient incurred 2 L of blood loss. She was transfused three units of packed cells perioperatively. Postoperatively, she had an uneventful recovery. The baby expired after 15 minutes due to extreme prematurity. The histopathology report of the sac tissue and right fallopian tube was collected which reported the presence of chorionic villi interspersed with blood vessels within the sac space while the tubal epithelium showed fibrin and blood clot within the lumen with no evidence of syncytiotrophoblast/villous structure (Fig. 3 ). Broad ligament pregnancy sac after extraction of fetus and placenta. Excised broad ligament tract after suturing. Histopathological examination slide showing the presence of chorionic villi interspersed with blood vessels within the sac space (staining done with hematoxylin and eosin, magnification 100x).

Ectopic pregnancy is an extrauterine pregnancy. Fallopian tube is the commonest site of ectopic pregnancy (95% cases) and abdominal pregnancy account for 1%. 1 Broad ligament pregnancy is defined as a gestation that grows in the space formed by anterior and posterior peritoneal folds of the broad ligament. It is an extrauterine pregnancy that is abdominal as well as extraperitoneal. The incidence is quoted as 1 in 10,000 to 1 in 30,000 pregnancies and 1% in ectopic pregnancies. 2 It poses a diagnostic dilemma and the diagnosis is seldom established before surgery and usually the obstetrician tackles the first broad ligament pregnancy of her life on operation table without prior preparations to face the challenge. It is a catastrophic obstetric emergency with a high rate of maternal mortality of about 20%. 3 Written consent for the use of case presentation and figures, and consent for its publication in print and electronically has been given by the patient.

Secondary broad ligament/intraligamentary pregnancy may result from tubal pregnancy or a uterine fistula. Rare cases are also reported after spontaneous dehiscence and separation of a previous cesarean section scar, after perforation of uterus during the process of therapeutic or elective termination of pregnancy, 4 or subsequent to a subtotal or total hysterectomy. Risk factors for broad ligament pregnancy include tubal abnormalities, previous salpingectomy, pelvic infection, endometriosis. Literature review shows that 29.4% of women with broad ligament pregnancy had previous history of any tubal surgery, majority having ipsilateral salpingectomy and approximately 12% had conceived from in vitro fertilization. 5 In our case, there was no such history or risk factor. The anatomical relation of the sac with the pelvic organs described by Champion and Tessitore is of paramount importance. 6 Superiorly, it is bounded by a fallopian tube, medial boundary formed by the uterus, laterally bordered by the lateral pelvic wall, and inferiorly bounded by the pelvic floor. The round ligament is commonly recognized prominently on the anterior surface of the gestational sac. It may have variable clinical presentations ranging from asymptomatic to hemodynamically unstable patients with a ruptured broad ligament pregnancy. The commonest complication reported was broad ligament pregnancy rupture, which was evident in 11 out of 30 cases of broad ligament or intraligamentary pregnancies which we reviewed during the literature search. Other causes of maternal morbidity include hemorrhage, sepsis, abscess, coagulopathy, and toxemia. The gestational age at presentation is reported to vary from 5 weeks 7 to 41 weeks with a few full-term cases reported till date. 8 – 13 Experiences with a broad ligament pregnancy vary with few being terminated at early gestation in view of hemodynamic unstability due to rupture and the advanced gestations progressing to term gestation with varying fetal outcomes. Few studies quote term broad ligament pregnancy with intrauterine fetal death, 14 few state term gestation with live born baby but culminating into an early neonatal death. 15 Term abdominal pregnancies with the favorable materno-fetal outcome resulting in live birth have been reported by few authors. 8 – 11 , 16 Rohilla et al. 17 reviewed 26 such cases resulting in live births at term. Clinical diagnosis is a dilemma and is usually made intraoperatively especially in advanced gestation. In literature search, we found some cases reported with a preoperative diagnosis, few opened for failed induction, some as elective cesarean sections, rest as tubal pregnancies. Here, in our case, we posted the case as a routine cesarean section. Azhar et al . 2 reported rupture of broad ligament pregnancy in a woman having a previous salpingectomy of ipsilateral tube. The investigation of choice is ultrasonography, trans-vaginal sonography is considered better than trans-abdominal ultrasound, where sac is identified adjacent to the inferior part of the uterus and an empty uterine cavity is identified. Broad ligament pregnancy can be easily missed in advanced pregnancy and in that case, Trans- abdominal ultrasound should be combined with trans-vaginal sonography to detect such cases. Magnetic resonance imaging gives an extra information regarding the extent of involvement of the uterus and mesentery and thus may aid in surgical planning. It may delineate the anatomical relationship between the placenta and area where the placenta has invaded, in order to be prepared preoperatively to face the challenge of massive blood loss and be armed with the availability of experienced obstetrician for stepwise devascularization, if needed. The mainstay of management is exploratory laparotomy with delivery of fetus and placenta and excision of the sac. Management dilemma remains for removal of the placenta which needs to be individualized. Site of placental insertion should be inspected carefully before removal as it may result in torrential hemorrhage due to injury to major vessels or may be buried in pelvic floor. In case reports reviewed, only one author reports to leave the placenta. If a patient in early gestation is hemodynamically stable, laparoscopic approach can be considered for excision of small-sized unruptured broad ligament preg-nancies. 18 , 19 Conservative or medical management is never recommended. Few studies also report an immediate laparotomy in early gestations in hemodynamically unstable patients. 20 , 21

Obstetrics is ever-changing and the rarest case can be encountered anytime, anywhere, even in low-risk pregnancies. So, it is very important to be thoughtful and watchful. Broad ligament pregnancy may be easily missed during antenatal period but a high index of clinical suspicion should be kept in cases of cervix deviated to one side with no obvious reasons, early-onset unexplained anhydramnios, and failed induction of labor. These all may raise high index of suspicion for an abdominal/broad ligament pregnancy. With reference to my case, I would comment that this index case had no risk factors mentioned above, so is probably a primary broad ligament pregnancy. There is no pathophysiology mentioned in detail in literature regarding oligoamnios, fetal anomaly, or intrauterine fetal death in broad ligament pregnancy. But, it can be considered scientifically that as the pregnancy develops in abnormal environment between the peritoneal folds of broad ligament instead of the thick uterine musculature, it must have been the cause for development of oligoamnios evident in my patient. The smooth muscles of broad ligament get distended with advancement of gestation and round ligament of corresponding side gets stretched over the growing sac leading to abdominal discomfort, pain, and tenderness and this was probably the cause for our distraction to consider it as uterine tenderness. Except oligoamnios, there was no factor to suspect this abnormal pregnancy. Non availability of an early pregnancy scan report was another hindrance to diagnosis. Repeated abdominal scans were done but if a transvaginal scan would have been done, we would have visualized uterine cavity and gestational sac separately. So, a case of unexplained oligoamnios, with no history and clinical examination findings supporting rupture of membranes, needs to be evaluated thoroughly to rule out extrauterine pregnancy even in advanced gestations. Rarity of the case, advanced gestation, and clinical symptoms supporting chorioamnionitis were hindrances to think out of the box for a broad ligament pregnancy and proceed to magnetic resonance imaging which would have given a preoperative diagnosis and a vision to face the battle armed with necessary arrangements.