Discussion
Ectopic pregnancy is a common emergency but life-threatening condition
that obstetricians and gynecologists face and requires fast and careful
management. In the past 30 years, the incidence of EP has raised in
developed countries [1]. Approximately 1% to 2% of all pregnancies
are ectopic pregnancies and over 98% of implantations occur in the
fallopian tube (7). The leading risk factors of EP in women are Pelvic
inflammatory disease, previous pelvic surgery, tubal infertility, and
congenital uterine abnormalities. (2, 3)
Bilateral tubal pregnancy (BTP) is considered an extremely rare form of
extra-uterine pregnancy [4]. It is difficult to estimate the
accurate frequency which is based on case reports, however, the highest
reported incidence was 1 in 200,000 pregnancies [5]. This
demonstrates 1/750 - 1,500 of all EPs [6]. BTP is commonly
associated with infertility treatment. Multiple ovulations either
spontaneously or with ovulation induction have elevated the risk of
bilateral EP [8]. In addition, the incidence has increased due to
the high rates of endo salpinx damages following sexually transmitted
infections, tubal sterilizations, assisted reproductive technologies,
tobacco smoking, polygamy, and more precise methods for early detection
of ectopic pregnancy. The highest risk factor for ectopic pregnancy is
previous fallopian tube damage [9, 10, 11].
Complications such as severe bleeding and hypovolemic shock associated
with maternal morbidity and mortality are the results of misdiagnosis or
delayed diagnosis of EP (2, 3). In this case, we report a patient with
bilateral tubal pregnancy, and the presentations were not at the same
time which makes it more life-threatening and harder to diagnose.
In cases of unilateral EP, early diagnosis can be successfully achieved
with the use of TVUS and the accessibility of β-hcg kits. Transvaginal
ultrasonography has a high sensitivity and specificity for detecting EP
and Color Doppler sonography increases the rate of transvaginal
ultrasound for early detection of small ectopic masses, preoperatively.
[7, 12] However, ultrasound has a weak role in the diagnosis of
bilateral EP, and almost all cases are diagnosed intraoperatively,
because even the most expert sonographers may encounter a handful in
their life career. [13] The literature review has shown it as an
operative diagnosis, except for only a few reports. Commonly, clinicians
fail to recognize the diagnosis during the surgery as well as our
patient. [12]
In our case, we didn’t visualize any abnormal findings on the
contralateral tube during the laparoscopic surgery. During the
post-operative follow-up due to the rise of β-hcg level and evaluating
other possibilities, we found out about the other ectopic pregnancy in
the contralateral tube. We consumed that during the laparoscopic
surgery, the other tubal pregnancy was too small to be seen then.
BTP treatment is controversial in most clinical guidelines for the
management of EP, Because of the rare cases of BTP [14]. Treatment
options for EP management are surgery, medical therapy, and expectant
management. Systemic MTX therapy is considered a cost-effective choice
rather than laparoscopy for patients who have stable hemodynamics.
Periodic β-hCG measurements are useful to diagnose EP and to assess the
efficacy of MTX. [15]
Clinicians should know about various therapeutical alternatives. Like
unilateral EP, choices depend on the patient’s condition, the extent of
damage to the fallopian tubes, the desire to preserve fertility, the
size and location of the EP mass, and the β-hCG level. [16]
Therapeutic options for spontaneous BTP cases are essentially similar to
unilateral EP. The type of surgical procedure may be different between
spontaneous BTP and those following ART. In spontaneous BTP, if the tube
appears benign, the procedure is to perform a salpingostomy. This is the
only successful pregnancy in the series [17].
Yao and Tolandi, [18] compared fertility rates between salpingotomy
versus salpingectomy, showing that both approaches were the same. Femke
et al [19] demonstrated a similar cumulative rate of natural
pregnancy among mentioned approaches.
Conservative management using methotrexate (MTX) does not satisfactory
therapeutic effect due to the high hCG level (the hCG concentration
level for MTX treatment was <5000IU/L). [20], salpingotomy
should be considered if the patient has a strong desire to save
fertility. However, patients should be aware of the possibility of
trophoblast tissue remnant and persistent trophoblast, Complementary
treatment with systemic MTX, and EP recurrence. [19, 21]
In our case, first, we did a laparoscopic salpingostomy and after the
diagnosis of BTP, we treated the patient with MTX therapy which needed a
second dosage.
Recent reports demonstrated delayed diagnosis of contralateral tubal
pregnancy days to weeks after the first surgery in BTP, so clinicians
should always keep in mind such an alternative, particularly, in
patients for whom multiple embryo transfers have been performed.
Bilateral fallopian tubes should always be checked, and inconsistent
β-hCG values may delay accurate diagnosis. [15] In our case, we were
able to diagnose it after the surgery and the rise of β-hCG level to
search for other possible locations of pregnancy.
Surgical procedures vary from salpingectomy for one tube and linear
salpingostomy for a contralateral tube to bilateral salpingostomy or
bilateral salpingectomy [21]. If present, laparoscopy may be the
best choice for diagnosis and management of BTP [22] because the
diagnosis can be easily missed even at laparoscopy so a high index of
suspicion should be kept. [22]. But in our case, as we inspected the
contra lateral tube thoroughly we didn’t observe any abnormal findings
due to the possibility of a small ectopic pregnancy in the right tube.
In hemodynamically unstable patients, laparotomy is the choice treatment
and is equally impressive. However, it should be noted that serial β-hCG
monitoring should be performed because there is a possibility of ongoing
ectopic pregnancy, especially if a conservative surgery such as
salpingostomy or tubal milking has been chosen [16]. There are
reports of intrauterine pregnancy after conservative surgical management
of BTP [23], but the reality is that these patients are at high risk
of recurrent ectopic pregnancy subsequently [16].
The unusual part of this case is the chronology of the bilateral ectopic
pregnancy. Most reported bilateral ectopic pregnancies occurred and
developed simultaneously, so the diagnoses were made at the same time on
both sides. In this case, even the mindful exploration of the
contralateral fallopian tube did not show any signs of a developing
ectopic pregnancy. At the first exploration, the contralateral implanted
embryo was too underdeveloped to be seen by laparoscopy, which
contributed to the consequently delayed diagnosis of ectopic pregnancy.
In this paper, we present an unusual case of bilateral ectopic
pregnancy, in which there has been a substantial delay of 2 weeks
between the diagnoses of both sides of the ectopic pregnancies.
Conclusion :
First of all, we should have this diagnosis in mind. Evaluation of both
adnexa by TVUS is equally important. A watchful inspection of the
abdomen and pelvis should always be performed during the surgery for an
ectopic pregnancy, particularly the contralateral fallopian tube even
when unilateral EP is diagnosed preoperatively. In some cases,
contra-lateral pregnancy was shown days to weeks after the first
surgery. This paper accentuates that diagnosis and treatment of one
ectopic pregnancy do not rule out the happening of a second ectopic
pregnancy in the same patient at the same time, especially if the
patient has rising β-hCG and persistent symptoms.
References :
1.Centers for Disease Control and Prevention (CDC). Ectopic pregnancy -
United States, 1990-1992. MMWR Morb Mortal Wkly Rep. 1995;44(3):46-48.
2. Jena SK, Singh S, Nayak M, Das L, Senapati S. Bilateral simultaneous
tubal ectopic pregnancy: a case report, review of literature and a
proposedmanagement algorithm. J Clin Diagn Res. 2016;10 (03):QD01–QD03.
Doi: 10.7860/JCDR/2016/16521.7416
3. Hortu I, Akman L, Akdemir A, Ergenoğlu M, Yeniel O, Şendağ F.
Management of ectopic pregnancy in unusual locations: five-year
experience ın a single center. J Clin Exp Invest.. 2017;8(03):90–95.
Doi: 10.5799/jcei.343197
4.Bouyer J, Coste J, Fernandez H, Pouly JL, Job-Spira N. Sites of
ectopic pregnancy: a 10 year population-based study of 1800 cases. Hum
Reprod. 2002;17(12):3224-3230.
5.Edelstein MC, Morgan MA. Bilateral simultaneous tubal pregnancy: case
report and review of the literature. Obstet Gynecol Surv.
1989;44(4):250-252.
6. Abrams RA, Kanter AE. Bilateral simultaneous extrauterine pregnancy.
Am J Obstet Gynecol. 1948;56(6):1198-1200.
7. Mayank M, Mohammed O, Andrew WH. Ectopic pregnancy. Obstet Gynecol
Rerod Med 2017;27:245–50.
8. Ayano F, Atsushi F, Rie F, et al. A case of bilateral tubal
pregnancy. Gynecol Minim Invasive Therapy 2017;6:191–2.
9. De Los Rios JF, Castaneda JD, Miryam A. Bilateral ectopic pregnancy.
J Minim Invasive Gynecol 2007;14:419–27.
10. Ankum WM, Mol BWJ, Van der Veen F, et al. Risk factors for ectopic
pregnancy: a meta-analysis. Fertil Steril 1996;65:1093–9.
11. K. T. Barnhart, M. D. Sammel, C. R. Gracia, J. Chittams, A. C.
Hummel, and A. Shaunik, “Risk factors for ectopic pregnancy in women
with symptomatic first-trimester pregnancies,” Fertilityand Sterility , vol. 86, no. 1, pp. 36–43, 2006.
12. J. Martinez, A. C. Cabistany, M. Gonzalez, O. Gil, M. Farrer, and J.
A. Romero, “Bilateral simultaneous ectopic pregnancy,” Southern
Medical Journal , vol. 102, no. 10, pp. 1055–1057, 2009.
13. Xu H. A spontaneous bilateral tubal pregnancy: A case report.
Medicine (Baltimore). 2018 Sep;97(38):e12365. doi:
10.1097/MD.0000000000012365. PMID: 30235696; PMCID: PMC6160232.
14. Barnhart MSCE, Franasiak JM. ACOG Practice Bulletin No. 191: tubal
ectopic pregnancy. Obstetrics Gynecology. 2018;131(2):409-411.
15. Acet F, Goker ENT, Hortu I, Sahin G, Tavmergen E. A Rare Case of
Bilateral Tubal Ectopic Pregnancy Following Intracytoplasmic Sperm
Injection-Embryo Transfer (ICSI-ET). Rev Bras Ginecol Obstet. 2020
Mar;42(3):165-168. doi: 10.1055/s-0040-1708093. Epub 2020 Mar 31. PMID:
32232825.
16. Andrews J, Farrell S, Andrews J. Spontaneous bilateral tubal
pregnancies: a case report. J Obstet Gynaecol Can. 2008;30(1):51-54.
17. Rani VRS, Puliyath G. Viable intrauterine pregnancy after
spontaneous bilateral tubal ectopics in a multiparous woman: a case
report. J Med Case Rep. 2013;7:159.
18.Yao M, Tulandi T. Current status of surgical and nonsurgical
management of ectopic pregnancy. Fertil Steril 1997;67:421–33.
19. Femke M, Annika S, Karin S, et al. Salpingotomy versus salpingectomy
in women with tubal pregnancy (ESEP study): an open-label multicenter
randomised controlled trial. Lancet 2014;383:1483–9.
20. Practice Committee of American Society for Reproductive Medicine-
Medical treatment of ectopic pregnancy. Fertil Steril 2008;90(5 suppl):
S206–12.
21. Van Mello NM, Mol F, Opmeer BC, et al. Salpingotomy or salpingectomy
in tubal ectopic pregnancy: what do women prefer? Reprod Biomed Online
2010;21:687–93.
22. Li W, Wang G, Lin T, Sun W. Misdiagnosis of bilateral tubal
pregnancy: a case report. J Med Case Rep . 2014;8:342.
23. Edelstein MC, Morgan MA. Bilateral simultaneous tubal pregnancy:
case report and review of the literature. Obstet Gynecol Surv.1989;44:250–52.