Intramural pregnancy managed by conservative laparoscopic resection: A case report.

The patient was a 30-year-old woman, gravida 7 para 3, + 2 ectopic pregnancies +1 M pregnancy. She had had in vitro fertilization with embryo transfer (IVF-ET) and was referred to the obstetrics department for an ultrasound scan on account of moderate vaginal bleeding at an estimated gestational age (EGA) of 8 weeks 6 days, associated with mild lower abdominal pain, with no trauma, vaginal instrumentation or use of abortifacient. The patient had history of a ruptured tubal ectopic pregnancy managed with left salpingectomy 7 years previously, and 2 years previously a right tubal ectopic pregnancy at the ampulla, which had been managed medically with methotrexate injection. She had also been diagnosed with molar pregnancy one year before this pregnancy. The molar pregnancy was diagnosed after histopathological assessment of tissues evacuated with a diagnostic dilation and curettage (D&C) of an unhealthy pregnancy with negative serum β-HCG titer for the 10 months that followed. The patient's medical history was unremarkable. Ultrasound at presentation revealed a gestational sac devoid of a fetal pole with a diameter of 2.7 cm, equivalent to 7 weeks of gestation, located high at the fundus adjacent to the superior margin of the endometrial plate on longitudinal scan and eccentrically located at the right cornual end on transverse scan. A diagnosis of right interstitial ectopic pregnancy was made. The patient was admitted for further workup. Her lab results after admission were: hemoglobin 121 g/L; platelets 215 × 10 9 /L; white blood cells 13.4 × 10 9 /L (with neutrophilia 81.5 %). Her liver function tests, kidney function tests, and coagulation profile were normal. B-HCG titer was 7600 mIU/mL. Therefore, the patient was prepared for laparoscopic exploration for suspected ruptured left interstitial pregnancy. Laparoscopy revealed the presence of a 2 cm ruptured uterus at the posterior upper uterine wall, as shown in Fig. 1 , with fetal tissue present, and a small amount of blood in the Douglas pouch. There was no connection between the ruptured site and the endometrial cavity, and so intramural ectopic pregnancy was suspected. Fig. 1 Laparoscopy revealed the presence of a 2 cm ruptured uterus at the posterior upper uterine wall. Fig. 1 Laparoscopy revealed the presence of a 2 cm ruptured uterus at the posterior upper uterine wall. Fetal tissue was removed from the rupture site and sent for histopathological examination along with an endometrial sample which was collected through D & C. After that, the rupture site was repaired with three separate stitches and suction of the hemoperitoneum was done. Also, a drain was retained as an extra precaution. The postoperative course was good. The drain had minimal output and was removed the subsequent day. The patient's postoperative hemoglobin level was 102 g/L. One week after her admission, the histopathology report stated that the ruptured site tissue was indeed fetal tissue, but the endometrial sample did not show any chorionic villi (as shown in Fig. 1 ). Therefore, the final diagnosis was ruptured intramural pregnancy.

Intramural pregnancy is a rare but possibly fatal condition. The reported case shows that multiple risk factors can co-exist in one patient. Diagnosis remains dependent of the expert's skill when using ultrasound. Laparoscopy is an efficient and safe method of managing intramural pregnancy and is a viable option for patients wanting to preserve fertility.

Due to the paucity of information in the literature, the aetiology and pathophysiology of intramural ectopic pregnancy remain unclear. When attempting to explain this unusual occurrence, two questions arise: how did the conceptus get into the myometrium and how did it get implanted? Three variables must be present for an intramural ectopic pregnancy to occur and for the conceptus to implant within the myometrium. The first and most frequently reported risk factor is the existence of a false tract that connects the myometrium and the endometrium. This typically results from prior uterine trauma in instrumentation procedures such as hysteroscopy or D&C, or from uterine surgery like myomectomy or caesarean delivery [ 6 ]. The second risk factor is cases of IVF-ET in which embryos are mistakenly transferred through a false tract that had been previously developed [ 6 ]. In such cases, embryos are inserted into the myometrium rather than the regular uterine cavity [ 6 ]. It is possible that in the case reported here, ET without ultrasound assistance increased the risk of intramural insertion. The third significant risk factor is adenomyosis, as it increases the myometrium's receptivity [ 7 ]. Adenomyosis appears to be the most plausible component in the development of intramural pregnancy, according to Lu et al. [ 7 ]. This is because there is enough endometrial tissue in deep adenomyosis to exhibit decidualization in response to oestrogen and progesterone, suggesting that such an area may be a suitable location for blastocyst implantation [ 7 ]. One or more of the risk factors listed above have been mentioned in prior case reports. The diagnosis of adenomyosis can be easily missed, particularly in the context of a gravid uterus, and hysteroscopy was not performed in some of these patients, meaning that the presence of a false tract could not be verified [ 8 ]. This case helps to explain the aetiology of intramural pregnancy by showing that all three predisposing factors—adenomyosis, IVF-ET, and false tract caused by prior surgery—can coexist. However, there are many cases of intramural ectopic pregnancy reported in the literature that did not display any risk factors [ 9 ]. Such cases may be explained by increased lytic activity of the syncytiotrophoblast and poor decidualization, which allow the conceptus to penetrate into the myometrium in a manner similar to placenta accreta [ 10 ]. Signs and symptoms of an ectopic pregnancy include pelvic pain and uterine hemorrhage along with a positive pregnancy test. Patients may not show any symptoms until they experience severe bleeding and uterine perforation, which may necessitate a hysterectomy and thus impair fertility [ 11 ]. Therefore, to avoid such radical measures, a timely and precise diagnosis is essential. One similar diagnosis is interstitial ectopic pregnancy, where the conceptus is implanted in the segment of the fallopian tube traversing the myometrium i.e. the interstitial segment [ 12 ]. To differentiate the two, it is essential to visualize the proximal segment of the interstitial tube adjoining the uterine cavity and the gestational sac [ 13 ]. The first-line diagnostic method is usually transvaginal ultrasound (TVUS), which usually reveals a fully encircled gestational sac with a “ring of fire” look on color Doppler imaging. Three-dimensional transvaginal ultrasound is superior because it provides improved visualization of the endometrium and myometrium independently and enables a more precise diagnosis of the gestational sac's placement within the myometrium using several scan planes [ 14 ]. Magnetic resonance imaging (MRI) is useful because it can better visualize the myometrium and endometrium independently and confirms the gestational sac's ectopic placement [ 13 ]. Memtsa et al. came to the conclusion that, in the majority of cases, a skilled ultrasound operator can make the diagnosis of intramural pregnancy without the need for further imaging, although some researchers have regarded it as the gold standard [ 13 ]. While the literature has not consistently reported the use of diagnostic hysteroscopy in cases of intramural ectopic pregnancy, it is a valuable tool that allows direct visualization of the uterine cavity and tubal ostium, and so can confirm the absence of the conceptus in the uterine cavity. In the case reported here, it also served another useful purpose by identifying the false tract and allowing visualization of the conceptus within its upper part. With the uterus preserved, the goal of conservative surgical care is to remove the ectopic pregnancy. This can be accomplished by laparotomy [ 15 ] or laparoscopy-assisted wedge resection or enucleation of the intramural ectopic pregnancy with myometrial repair [ 16 ]. Laparoscopic incision, aspiration, and interrupted sutures were described by Park et al. [ 17 ]. When an ectopic pregnancy bulges through the serosal surface, which happens in a more advanced gestation, this method might be appropriate. The increased morbidity of surgical management is one of its disadvantages. In addition, the myometrium must be rebuilt, which increases the possibility of a rupture in subsequent pregnancies.

Only about 70 cases of intramural ectopic pregnancy are documented in the literature. It accounts for less than 1 % of ectopic pregnancies [ 1 ]. When the gestational sac is completely inside the myometrium, apart from the endometrial cavity and fallopian tubes, this condition is known as intramural ectopic pregnancy, and is potentially fatal [ 2 ]. The symptoms of amenorrhea, lower abdomen pain, and vaginal bleeding that accompany intramural ectopic pregnancy are non-specific and can mirror the symptoms of other types of ectopic pregnancy [ 1 ], leading to occasional misdiagnosis of an intramural ectopic pregnancy as myoma or choriocarcinoma [ 3 ]. Furthermore, in some of the reported cases of intramural ectopic pregnancies, patients reported little discomfort [ 4 ], and in one case the blood β-Human chorionic gonadotropin (β-HCG) was even null [ 5 ]. Because intramural ectopic pregnancy is uncommon, there is a dearth of information on the clinical features, available treatments, and long-term reproductive outcomes of patients; most of the information that is available comes from case reports. We describe here the case of a 30-year-old woman experiencing an intramural ectopic pregnancy who was treated with laparoscopic conservative excision of the ectopic mass.