Spontaneous Unilateral Tubal Twin Ectopic Pregnancy

Healthcare providers should have a high index of clinical suspicion for ectopic pregnancies. Unilateral tubal twin ectopic pregnancies, while rare, can be adequately diagnosed with transvaginal ultrasound. Laparoscopic salpingostomy pro- vides conservative and successful treatment of unilateral tubal twin gestation with short recovery time. Medical treatment with methotrexate has not been adequately studied in this clinical scenario, but further exploration of this management modality shouldbeconsidered.

Pregnancy–ectopic, pregnancy–tubal, twin ectopic Address correspondence to Veronica C. Gillispie, MD, FACOG, Department of Obstetrics and Gynecology, Ochsner Clinic Foundation, Ochsner Baptist Medical Center, 4429 Clara Street, Suite 500, New Orleans, LA 70115. Tel: (504) 842-9617. Email: [email protected]

Ectopic pregnancy accounts for approximately 1%-2% of first-trimester pregnancies and may be the cause of up to 6% of pregnancy-related deaths.1 Risk factors for an ectopic pregnancy include prior tubal surgery, pelvic inflammatorydisorder,peritubaladhesions(followingappen- dicitis or endometriosis), congenital tubal anomalies (ie, in utero diethylstilbestrol exposure), artificial reproductive ther- apy, and smoking. Although contraceptive methods over- all decrease the risk of ectopic pregnancy by preventing all pregnancies, intrauterine devices (IUDs) may actu- ally increase the risk. 1 Early detection of ectopic preg- nancy has improved via urine/serum beta-human chorionic gonadotropin (bHCG) testing and transvaginal ultrasound, thus decreasing associated morbidity and mortality. Tubal ectopic pregnancies comprise 95% of ectopic pregnan- cies. Within this incidence lies the even rarer occurrence of tubal twin ectopic pregnancy, with incidence rates esti- mated to be 1 in 725-1,580 of tubal pregnancies. 2 Unilat- eraltubaltwinpregnanciesoccurinapproximately1ofevery 125,000 spontaneous pregnancies. 3 A diagnosis of prior ectopic pregnancy increases the risk of subsequent ectopic pregnancy by 10%.1 Management options for ectopic pregnancy are medi- cal management via methotrexate or surgical management. Because of the paucity of data, little guidance for the appro- priate management of a twin tubal ectopic pregnancy is available. CASE REPORT A40-year-oldfemalegravida1para1001presentedtothe emergency department (ED) with a 2-day history of vaginal bleeding associated with lower abdominal pain. The patient stated that she had filled several pads with dark red blood andlargeclotsthatlookedlike“tissue.”Shedeniedanyvagi- nal pain or discharge, dysuria, and frequency or urgency of urination. The patient also denied constitutional symp- toms, as well as gastrointestinal symptoms. Her last men- strual period was 6 weeks prior to presentation. She denied any history of sexually transmitted disease. She had no sig- nificant medical history, and her surgical history was only significant for cesarean section for her first child (17 years previously). The patient denied taking any medications and is a nonsmoker. Onphysicalexamination,thepatientwasafebrilewithsta- ble vital signs. On abdominal examination, the patient was nontender, soft, and nondistended with no signs of rebound tendernessorguarding.Examinationoftheexternalgenitalia showednoabnormalities.Pelvicexaminationrevealedsome darkbrownbloodinthevaginalvaultwithnoactivebleeding. 178 OchsnerJournal Lategan, HE Figure. A: Transvaginal ultrasound sagittal view shows the right fallopian tube containing 2 discrete gestational sacs. B: Transvaginal ultrasound transverse view shows the right fallopian tube containing 2 discrete gestational sacs. The patient had no cervical motion tenderness. Bimanual examination revealed a uterus of 6-week size with a closed cervical os. No adnexal masses were present; however, the patient acknowledged tenderness on palpation of the right adnexa. In the ED, a urine pregnancy test was positive. Blood work showed serum bHCG of 23,359 mIU/mL, hemoglobin of 10.4 g/dL, hematocrit of 32.4%, and O positive blood type.Transvaginalultrasound(Figure)revealedauterusmea- suring 8.3 cm in length and 3.9 × 4 . 5c mi nt r a n s v e r s e dimensions with no intrauterine gestational sac; however, 2 closely opposed gestational sacs, each containing a fetal pole, were seen in the right adnexal region separate from the ovary. The left ovary was not identified, and no left adnexal abnormalities were seen. Fetal heart tones were not detected at either fetal pole. No significant amount of free fluid was present within the pelvis. Overall, these find- ings indicated a right tubal ectopic twin pregnancy of aver- age ages of 8 weeks and 2 days and 7 weeks and 6 days, respectively. The patient was emergently taken to the operating room for diagnostic laparoscopy with right salpingostomy. Intra- operative findings were consistent with the preoperative diagnosis. The patient’s postoperative course was uncom- plicated,andshewasdischargedonpostoperativedayzero. Histopathology confirmed the diagnosis. Serial bHCG levels were checked until the level was<5m I U / m L .

From the report in 1891 by De Ott4 through 1990, only 98 cases of unilateral tubal twin pregnancy had been described in the literature, most of which were diagnosed intraop- eratively or postoperatively. 5 The 101st case of unilateral twin ectopic pregnancy was described only in 2001.6 In 2006, Rolle et al reported that only 8 cases of unilat- eral tubal twin pregnancy in the literature had been diag- nosed preoperatively. 7 In the current case, the treatment team was able to establish with reasonable certainty that the patient had a unilateral twin ectopic pregnancy prior to surgery. The literature includes case reports describing twin ectopic gestations following salpingectomies and other tubalsurgeries,aswellasothersfollowingahistoryofpelvic infection or artificial reproductive therapy. This patient, how- ever, had no history of tubal surgery, pelvic infection, artifi- cial insemination, tubal anomalies, IUDs, or smoking. In all women of reproductive age, even those with no known risk factors, physicians should operate with high clinical suspi- cion of potential ectopic pregnancy. Because of the low incidence of spontaneous unilateral tubal twin pregnancies, no consensus has been reached as to the best option for management. In cases similar to ours, laparotomyandlaparoscopicsalpingectomyhavebeensuc- cessfully performed. 8 Only one other case report describes management via laparoscopic salpingostomy as achieved in our case. Only 2 cases of medical management with methotrexate have been documented in hemodynamically stablepatientswithunrupturedunilateraltubaltwinpregnan- cies, and both were successful. 9,10

Even in the absence of the accepted risk factors, the possibility of unilateral twin ectopic pregnancy should be considered. Retrospectively in this case, treatment with methotrexate may have been an option; however, with extremely limited data on the use of medical management, methotrexatemaynothave beenappropriate,but thisques- tion is an imperative for future study. ACKNOWLEDGMENTS The authors have no financial or proprietary interest in the subject matter of this article. Volume19,Number2,Summer2019 179 Spontaneous Unilateral Tubal Twin Ectopic Pregnancy

1. CunninghamFG,LevenoKJ,BloomSL,etal.Ectopic pregnancy.In:CunninghamFG,LevenoKJ,BloomSL,etal, eds. Williams Obstetrics.24thed.NewYork,NY:McGraw-Hill Education;2014:377-395. 2. SvirskyR,MaymonR,VakninZ,etal.Twintubalpregnancy:a risingcomplication? Fertil Steril.2010Oct;94(5): 1910.e13-1910.e16.doi:10.1016/j.fertnstert.2010.03.017. 3. DedeM,GezginçK,YenenM,etal.Unilateraltubalectopic twinpregnancy. Taiwan J Obstet Gynecol.2008 Jun;47(2):226-228.doi: 10.1016/S1028-4559(08)60089-3. 4. DeOttD.Acaseofunilateraltubaltwingestation. Ann Gynécol Obstét.1891;36:304. 5. ShererDM,LibertoL,WoodsJRJr.Preoperativesonographic diagnosisofaunilateraltubaltwingestationwith documentedfetalheartactivity. J Ultrasound Med.1990 Dec;9(12):729-731. 6. GökerEN,T avmergenE,OzçakirHT,LeviR,AdakanS. UnilateralectopictwinpregnancyfollowinganIVFcycle. J Obstet Gynaecol Res.2001Aug;27(4):213-215. 7. RolleCJ,WaiCY,BawdonR,Santos-RamosR,HoffmanB. Unilateraltwinectopicpregnancyinapatientwithahistory ofmultiplesexuallytransmittedinfections. Infect Dis Obstet Gynecol.2006;2006:10306. 8. SurSD,ReddyK.Spontaneousunilateraltubaltwin pregnancy. JRS o cM e d.2005Jun;98(6):276. 9. KaradenizRS,DilbazS,OzkanSD.Unilateraltwintubal pregnancysuccessfullytreatedwithmethotrexate. Int J Gynaecol Obstet.2008Aug;102(2):171. doi:10.1016/j.ijgo.2008.03.012. 10. ArikanDC,KiranG,CoskunA,KostuB.Unilateraltubaltwin ectopicpregnancytreatedwithsingle-dosemethotrexate. Arch Gynecol Obstet.2011Feb;283(2):397-399. doi:10.1007/s00404-010-1449-6. This article meets the Accreditation Council for Graduate Medical Education and the American Board of Medical Specialties Maintenance of Certification competencies for Patient Care and Medical Knowledge. 180 OchsnerJournal