Abstract
Introduction:
Ectopic pregnancies complicate 11/1000 pregnancies in the United Kingdom and contribute significantly to maternal morbidities and mortality. Twin ectopic pregnancies are rare and preoperative diagnosis extremely so. The risk factors and clinical presentation for twin ectopic pregnancies are the same as for ectopic pregnancies in general. The nature of twin ectopic pregnancies, however, implies a potential for higher complication rates. Majority of cases are diagnosed and thus managed surgically; however, successful medical management has been documented with preoperative ultrasound diagnosis.
Case Presentation:
A nulliparous patient presented at six weeks gestation with abdominal pain and vaginal bleeding. An ultrasound diagnosis of right tubal twin ectopic pregnancy was made. She was initially commenced on medical management, but this was abandoned due to worsening symptoms. She subsequently had a laparoscopic right salpingectomy, which was uncomplicated.
Discussion:
The case presented was one of unilateral twin tubal ectopic pregnancy, which is a rare occurrence. In this case, the diagnosis was preoperative unlike most reported cases. Although some reported cases have been successfully managed medically, medical management was unsuccessful in this case, necessitating surgical management.
Conclusion:
Current guidelines for management of ectopic pregnancies do not consider twin ectopic pregnancy, and hence, there is presently no guidance as to the preferred management options for this unique subset of patients. Medical and surgical management options have been utilized successfully in reported cases. While most reported cases were diagnosed and managed surgically, medical management remains an option in carefully selected cases.
Keywords
Introduction
Ectopic pregnancies are a major contributor to maternal mortality globally. In the United Kingdom, the incidence is 11/1000, accounting for 12,000 ectopic pregnancies annually.1,2 In the period from 2021 to 2022, the 12 maternal deaths that occurred from early pregnancy causes were all due to ectopic pregnancies. 1 Of these deaths, approximately 75% could have been avoided, hence the importance of a good understanding of the condition. 1
An ectopic pregnancy is a pregnancy in which the fertilized oocyte is implanted outside the normal endometrial cavity. This implantation may occur within the Fallopian tubes, the ovaries, the cervix, caesarean section scar and the abdominal cavity. Fallopian tubes tend to be the commonest site for abnormal pregnancy implantation, accounting for more than 90% of all ectopic pregnancies. Other sites are less common 2 . Rarely, multiple pregnancies like other pregnancies could be abnormally implanted. This could involve an intrauterine pregnancy co-existing with an ectopic pregnancy, known as a heterotopic pregnancy, or with multiple extrauterine pregnancies, which could be unilateral or bilateral tubal pregnancies. The reported incidence of unilateral twin ectopic pregnancies is 1:20,000 to 1:250,000. 3 Since reporting began in 1986 until December 2023, 106 cases have been reported.4,5 Only a handful of these cases (under 10%) have been diagnosed preoperatively. 5 Here, we present a case of unilateral twin ectopic pregnancy diagnosed preoperatively by ultrasound.
The risk factors for twin ectopic pregnancy are the same as for ectopic pregnancies in general and include previous ectopic pregnancies, tubal damage from infections, inflammation or previous surgery, smoking, increased maternal age, subfertility and its treatment including in vitro fertilization.2,6,7 Of particular note is the increased risk of twin gestation in particular ethnic groups and following assisted fertility. 8 A third of patients have no identifiable risk factors before the diagnosis of an ectopic pregnancy. 2 The diagnosis of twin ectopic pregnancies is similar to that of singleton ectopic pregnancies and is usually based on clinical features and ultrasound findings which include the absence of an intrauterine pregnancy and other features that may include the following:
The finding of multiple gestational sacs.
Complex adnexal masses.
Pseudointrauterine sac.
Free pelvic fluid. 5
Twin ectopic pregnancies have been managed medically with the use of methotrexate administered in single or multiple doses and surgically by salpingectomy and salpingostomy.4,5,9 Of the six reported cases of medical management, four were successful, that is did not require further surgical treatment. 3 It should, however, be noted that majority of reported cases were diagnosed at surgery and the implication of this is potential underreporting of cases which would have been managed expectantly or medically and never diagnosed as twin ectopic pregnancies. 4 This underscores the importance of preoperative ultrasound diagnosis.
Case presentation
A 23-year-old nulliparous woman presented at approximately six weeks of gestation with abdominal pain and vaginal bleeding. Abdominal pain began a week before presenting but became significantly worse the day before attending Emergency department. The pain had evolved from a diffuse lower abdominal pain to a localized right lower abdominal pain. Vaginal bleeding was described as spotting. This pregnancy was a planned spontaneous conception. She had previously had a spontaneous first-trimester miscarriage. She smoked but had no previous abdominal or pelvic surgeries. At the initial examination, the patient was clinically stable, and her abdomen was soft with suprapubic and right iliac fossa tenderness but no guarding. The cervical os was closed with no demonstrable vaginal bleeding. Initial blood investigations included a haemoglobin concentration (Hb) of 127 g/L, platelet count of 316/L and serum beta hCG of 1652 mUI/mL. Transvaginal ultrasound scan performed on the same day showed the presence of a 22 mm × 19 mm × 25 mm mass, medial to and separated from the right ovary, containing two irregularly shaped gestational sacs, each containing an embryonic pole, neither demonstrating cardiac activity. They had a crown rump length measurement of 2 and 1.6 mm, respectively, which put gestational age at approximately five weeks. The uterus had an endometrial thickness of 9.5 mm with no evidence of an intrauterine pregnancy. Both ovaries appeared normal, and there was no free pelvic fluid seen (Images 1 to 8).
Longitudinal section of the uterus (transabdominal).
Longitudinal section of the uterus (transvaginal).
Transverse section of uterus (transvaginal).
Right adnexa, showing right ovary adnexal mass containing two gestational sacs.
Zoomed out image of adnexal mass with both gestational sacs with one embryonic pole.
Zoomed out image focusing on second gestational sac with embryonic pole.
Transverse section of left adnexa showing left ovary.
Longitudinal section of left adnexa.
The diagnosis at this point was a unilateral twin ectopic pregnancy. In view of the size, the absence of fetal cardiac activity or evidence of rupture and the hemodynamic stability, the patient was offered management options that included medical treatment using methotrexate and surgical treatment with laparoscopic salpingostomy or salpingectomy. She opted for medical treatment and had an intramuscular dose of methotrexate on the same day and remained an in-patient in view of an ongoing complaint of pain.
On the first day of admission, her abdominal pain and tenderness persisted, with associated nausea. She, however, was keen to continue with medical management. Her vitals remained within normal limits.
On the second day of admission, her pain and tenderness were significantly worse; however, her vital signs remained within normal limits, and a repeat full blood count showed a stable haemoglobin concentration. In view of her symptoms, the management options were re-discussed and this time she opted to have surgical management and gave her consent for the same (Images 9 and 10).
Grossly distended tube, with bleeding from the fimbrial end.
Post total right salpingectomy.
She had a laparoscopy, and the findings included the following:
Blood and tissue removed from the cervical os which was otherwise closed.
Approximately 200 mL of haemoperitoneum, right fallopian tube distended from isthmus to fimbriae with bleeding from the fimbrial end.
Normal left fallopian tube.
Normal ovaries and normal uterine appearance.
No pelvic or intra-abdominal adhesions.
Laparoscopic Images
Right total salpingectomy was performed, and the haemoperitoneum was washed out. Surgery was uncomplicated, and she was discharged on the following day, having recovered well.
Histology confirmed the diagnosis of tubal ectopic pregnancy with the presence of trophoblastic tissue; however, there was no definite fetal tissue. The tissue that was removed from the cervix was confirmed to be a decidual cast.
Discussion
The case presented is one of unilateral twin ectopic pregnancy diagnosed preoperatively by ultrasound scan and managed surgically. Preoperative diagnosis of twin ectopic pregnancy is a rare find with only under 10% of the 106 reported cases being diagnosed preoperatively. 5 A high index of suspicion of ectopic pregnancy should be maintained in reproductive aged females who present with abdominal pain and vaginal bleeding; however, in many cases, the symptoms are vague, and hence, a pregnancy test should be part of initial investigations for reproductive aged females attending the emergency department. 1 The index of suspicion should be higher in the presence of risk factors for ectopic pregnancies which include previous ectopic pregnancy, tubal damage, infertility and treatment, and smoking. 2 In the reported case, the patient attended with symptoms of abdominal pain and vaginal bleeding. She also had a known risk factor of being a smoker. Smoking has been shown to alter ciliary function within the fallopian tube, hence altering trans-tubal transportation of the fertilized ova, and thus increasing the likelihood of ectopic pregnancy. 7
The majority of ectopic pregnancies are diagnosed on the initial ultrasound scan 2 as in this case. It should, however, be noted that although transvaginal ultrasound scans have a sensitivity and specificity of diagnosing tubal ectopic pregnancy of 87.99% and 94% to 99.9%, respectively, 2 the ultrasound features make it unlikely to objectively diagnose twin ectopic pregnancies. These features include the absence of an intrauterine gestational sac, a complex adnexal mass that moves separately to the ovary, a gestational sac and rarely fetal cardiac activity. This likely provides an explanation for the rarity of preoperative diagnosis and possible under reporting of twin ectopic pregnancies. In this case, however, the diagnosis was made on the initial ultrasound scan as an adnexal mass that was separate to the ipsilateral ovary and containing two gestational sacs.
Most reported cases of twin ectopic pregnancies were managed surgically. This is likely due to the timing and nature of diagnosis. Juneau and Bates 10 have demonstrated no significant difference in success rates between expectant, medical and surgical management of singleton ectopic pregnancies among well-selected patients. Applying similar selection criteria as for singleton ectopic pregnancies may enable safe trial of expectant or medical management of twin ectopic pregnancies, and therefore avoiding the potential morbidity associated with surgery and affording patients the opportunity to be involved in their management by deciding on their preferred management option. Of the six reported cases of medically managed twin ectopic pregnancies, four were successful, that is not requiring further surgical management.4,9 The National Institute of Health and Care Excellence (NICE) and Royal College of Obstetricians and Gynaecologists have recommended selection criteria for the use of methotrexate in the management of ectopic pregnancies. These criteria include the patient being able to attend for follow-up, absence of significant pain, absence of evidence of rupture, under 35 mm size and no viable heartbeat, a serum hCG level of between 1500 and 5000 and the absence of an intrauterine pregnancy.2,7 In the managed case, all the above selection criteria were met. The patient, however, had pain as a symptom but was keen to avoid having surgery. The medical management was eventually abandoned for surgery due to persistent and worsening pain. At surgery, a unilateral salpingectomy was performed as the contralateral fallopian tube was found to be normal. This is the recommended preferred procedure over a salpingostomy which preserves the affected tube. This preference is based on the lower incidence of persistent trophoblastic tissue and improved future reproductive function. 2
Conclusion
The case presented was of unilateral twin tubal ectopic pregnancy. Initial medical management was unsuccessful, and the pregnancy was subsequently managed surgically by laparoscopic unilateral salpingectomy. While medical management remains a reasonable option for management of twin tubal ectopic pregnancies, it is important to carefully select patients within established recommendations. Although these recommendations do not presently consider twin ectopic pregnancies specifically, adherence to the guidance is an important first step in patient selection. The ability to carefully select cases and offer options for management is dependent on being able to make a preoperative diagnosis by ultrasound scan in a patient that is otherwise hemodynamically stable.
Footnotes
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