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Iran J Reprod Med Vol. 12. No. 12. pp: 825-830, December 2014 Original article

Ovarian ectopic : A 10 years’ experience and review of literature

Lajya Devi Goyal1 M.D., Rimpy Tondon2 M.D., Poonam Goel2 M.D., Alka Sehgal2 M.D.

1. Department of and Abstract Gynecology, Guru Gobind Singh Medical College and Hospital, Background: Primary ovarian pregnancy is one of the rarest forms of ectopic Faridkot, Panjab, India. pregnancy having incidence of 1/7000-1/40,000 in live births and 0.5-3% of all 2. Department of Obstetrics and ectopic gestations. Intrauterine contraceptive device (IUCD), salpingitis, infertility, Gynecology, Govt. Medical and assisted reproductive techniques are the important risk factors. Approximately, College, Chandigarh, India. 75% terminate in first trimester and are often misdiagnosed as haemorrhage. Preoperative diagnosis by ultrasonography (USG) in early pregnancy can help in conservative medical/ surgical management. Objective: The aim of the present study was to find the incidence, risk factors, role

of USG in pre-operative diagnosis, feasibility of conservative management with medical method or minimal invasive surgery in developing countries like India. Materials and Methods: We did a retrospective cross-sectional study of ovarian managed at Government Medical College and Hospital Chandigarh

between July 2000 to July 2010. We analyzed the incidence, risk factors, clinical presentation, management of ovarian pregnancy, and reviewed the literature. Results: Incidence of ovarian pregnancy was 4.9% of all ectopic pregnancies (14/523). Thirteen (93%) patients presented in first trimester with acute pain

abdomen and of these ten patients had bleeding per vaginum. One (7%) patient referred from peripheral hospital at term gestation with ultrasonographic diagnosis of breech presention with plecenta previa. Pre-operative diagnosis was made only in

two cases (11%). All cases were managed by . Excision of the sac with Corresponding Author: conservation of the was done in eleven cases (78%) and was Lajya Devi Goyal, Department of done in two cases (14%). Obstetrics and Gynecology, Guru Conclusion: Incidence of ovarian pregnancy is on the rise. Although Gobind Singh Medical College and Hospital, Faridkot, Panjab, India. ultrasonography can detect ovarian gestations in unruptured cases but cannot easily Email: [email protected] differentiate ovarian from other tubal gestation in ruptured state. Medical Tel: (+91) 941 7658846 management is usually not feasible it most of the patients present in ruptured state. Conservative surgical approach is the management of choice. Received: 21 October 2013 Revised: 5 July 2014 Accepted: 17 August 2014 Key words: , Ovarian pregnancy, Infertility.

Introduction ovarian pregnancy. In advance pregnancies last criterion i.e. detection of ovarian tissue in varian pregnancy is uncommon the wall of sac may not be satisfied as form of ectopic pregnancy with an parenchyma is compressed laminated and O incidence of 1/7000-1/40,000 live distended by developing foetus (3). births and 0.5-3% of all ectopic gestations (1). The actual incidence could be up to 1 in Advance ovarian pregnancies are exceptional. 1400 deliveries if the criteria other than those Approximately 75% terminate in first trimester of Spielberg are taken into consideration. and are often misdiagnosed as corpus luteum These criteria combine biochemical and USG haemorrhage (2). The Spielberg criteria findings and include a) serum βhCG level (1878)- a) as the affected site ≥1000 IU/L b) no in uterine at must be intact b) the foetal sac must occupy transvaginal ultrasound c) ovarian the position of the ovary c) the ovary must be involvement should be confirmed on connected to the by exploration, with bleeding, visualization of and d) ovarian tissue must be located in the chorionic villi or presence of atypical cyst as sac wall, are essential for confirmation of early the ovary d) normal tubes e) absence of Devi Goyal et al serum βhCG after treatment of ovary (4). gestations fourteen were ovarian, four Intrauterine contraceptive device (IUCD) use cervical, three abdominal and 502 were tubal was the only risk factor quoted in the past gestation. All patients fulfilled Speigelberg’s literature and salpingitis and infertility are not criteria. One case of ovarian pregnancy implicated but recent literature quote presented at term (10). The mean age of the increased incidence with infertility and patients was 27 years. Mean gestational age assisted reproductive techniques (5-7). was 42 days. Eight patients presented with Role of USG in diagnosis of ovarian amenorrhea. One patient presented at term gestation has been described but most of the gestation and in five patients there was no patients present with ruptured ectopic and are history of amenorrhea. Most common in circulatory collapse so preoperative presenting complaint was acute pain diagnosis of ovarian ectopic on sonography is abdomen in 13 (93%) patients followed by not easy (8, 9). We conducted a retrospective in 10 (71%) patients. Six analysis and reviewed the case records of patients showed evidence of circulatory fourteen ovarian pregnancies including one collapse at presentation. Vaginal findings case of term ovarian pregnancy among 523 showed ectopic pregnancy in eleven patients. ectopic gestations and 33285 deliveries. There was history of IUCD use in five cases The aim of the present study was to find the incidence, risk factors, possibility of pre- (duration 1-6 years). operative USG diagnosis, feasibility of History of infertility was present in two conservative management with medical cases, of these one patient had undergone method or minimal invasive surgery. induction and intra uterine insemination in the preceding cycle. History of Materials and methods spontaneous or induced was present in five cases and six patients were nullipara A retrospective cross-sectional study of (three were primigravida and three had ovarian pregnancies was conducted in previous one or two first trimester ). department of Obstetrics and Gynecology in Pregnancy test was positive in thirteen our Institute over a period of ten years i.e. July patients (pregnancy test was not done in case 2000-July 2010. The case records of ovarian of term ovarian pregnancy). βhCG test was pregnancies were searched in central record done in seven patients only (range: 1600- department of the hospital after taking 3200 IU/L) as other patients could not afford permission from medical record section to due to financial constraints. review the records of all those patients USG findings revealed echogenic sac with diagnosed with ovarian pregnancy for fetal cardiac activity in the ovary only in two academic and research purpose. cases. Adnexal mass was present on USG in Verbal consent was taken telephonically five cases varying from 4x4cm to 7x6cm. Rest from all the patients. Baseline information, of the cases showed free fluid in peritoneal presenting features, investigations and cavity with clots in pouch of doughlas. Term operative details were recorded. There is no ovarian pregnancy was misdiagnosed as control group in this study, because it breech presentation with placenta previa on investigates and reports the present files. USG. All patients underwent laparotomy. Intra operative findings revealed sac in the process Results of extrusion and surface bleeding from right ovary in nine cases. There were 523 ectopic pregnancies Term ovarian pregnancy was also in the among 33285 deliveries (1.7%). In ectopic right sided ovary. Left ovarian pregnancy

826 Iranian Journal of Reproductive Medicine Vol. 12. No. 12. pp: 825-830, December 2014 Ovarian pregnancy presented as an adnexal mass in two cases cases. Oophrectomy was done in two and ruptured cystic mass in two cases. Intra cases as sac was deep seated and operative diagnosis of corpus luteal bleeding could not be controlled. In term hemorrhage was suspected in three cases ovarian pregnancy entire ovary was however ovarian pregnancy were confirmed incorporated in sac so excision of the sac by histopathology examination in all these was done. On follow up pregnancy test cases. Excision of the sac with was negative in all cases of early ovarian conservation of the ovary was done in 11 pregnancies (Table I).

Table I. Age, parity, risk factor, diagnosis and treatment of ovarian pregnancy Period of Serial Age Ultrasonography Intraoperative Parity Gestation Risk Factors Treatment number (years) Findings/ β-hCG findings (weeks) Ecogenic Sac with sac extruding Excision of sac B/L 1 24 G P L A 6+2 CuT-2yr 5 2 2 2 FCA in R adnexa from R ovary salpingectomy Primary infertility Nil sac extruding 2 22 Nulliparous ovulation induction, FF in peritoneal cavity Excision of sac from L ovary IUI sac in R ovary with Excision of sac B/L 3 26 G P L A 6 CuT-1yr FCA G sac in R ovary 4 2 2 1 salpingectomy βhCG -2200iu/l 5×5 cm L adnexal L ovary embedded 4 28 P L Nil CuT-3yrs L Oophrectomy 3 3 mass, echogenic area in clots MTP in early Placenta previa, pregnancy Live 2.2 kg 5 24 G P L 40 Breech, Excision of sac 2 1 1 Continuation of in R ovarian sac oligohydraminos pregnancy FF in peritoneal cavity R ovarian 2cm 6 35 G P L 6+1 Nil Excision of cyst 2 1 1 βhCG 1880iu/l mass R adnexal mass Protruding sac 7 24 Primi 6+1 Nil Excision of sac FF from R ovary R ovarian sac with 8 35 G3P2 L2 5 CuT-6yr fetal node. βhCG R ovarian sac Excision of sac 2400iu/l 1.5 cm rent in R 9 24 Primi 6+2 Nil FF in abdomen Excision of cyst ovary

L adnexal mass, βhCG 5×5 cm L ovarian Excision of cycst 10 26 P L Nil Nil 3 3 -3200iu/l ruptured mass and repair

L adnexal mass βhCG Ruptured sac in L 11 32 Nulliparous Nil Nil Excision of sac 1800iu/l Ovary

R adnexal mass, βhCG 5×4cm R ovarian 12 26 G P L A 7+2 CuT-2yr Excision of sac 5 1 1 3 -3000iu/l Cyst

Sac extruding R 13 20 G A 5+2 Nil FF in abdomen Excision of sac 2 1 ovary

R adnexal mass βhCG- 14 32 Nulliparous Nil Infertility Sac in R ovary R Oophrectomy 2400iu/l FCA:Fetal cardiac activity R: Right L: Left FF: free fluid B/L: bilateral

Iranian Journal of Reproductive Medicine Vol. 12. No. 12. pp: 823-828, December 2014 827 Devi Goyal et al

Discussion rupture in the early first trimester and are usually misdiagnosed as corpus luteal Although ovarian pregnancy is a rare entity haemorrhage (2). Advanced ovarian but incidence is on rise. Quoted incidence in pregnancies are exceptional (in our series past were 0.5-1% of all ectopic gestation or 1 term ovarian pregnancy with live birth has in 7,000-40,000 live births (1). In our series been reported). Term healthy live birth out of incidence of ovarian pregnancy were 1.7% of ovarian pregnancy is very rare. Although all ectopic and 1 in 3332 deliveries which advanced ovarian pregnancies with almost was doubled. Recent studies suggest intrauterine death have been reported (3). that the actual incidence could be up to 1 in Classical triad of ectopic pregnancy i.e. 1400 deliveries if the criteria other than those amenorrhoea, pain abdomen and vaginal of Speigelberg’s are taken into consideration bleeding was present only in six cases. Role (4). of USG in diagnosis of ovarian gestation has There is overall increase incidence of been described by various authors. The ectopic gestation due to increasing prevalence following sonographic diagnostic criteria for of sexually transmitted disease and PID, the presence of an ovarian pregnancy have induced abortions, assistant reproductive been suggested: a wide echogenic ring with techniques and increased availability of an internal echo lucent area on the ovarian diagnostic facilities. Increased incidence of surface; the presence of ovarian cortex, ovarian pregnancies may be associated with including corpus luteum or follicles around the IUCD as it prevents intrauterine but not extra mass; and the echogenicity of the ring usually uterine pregnancies. It is postulated that IUCD greater than that of the ovary itself (8, 9). may potentiate ovarian nidation due to Recently, a case was diagnosed with 3- changes in synthesis of prostaglandin which dimensional sonography (13). increase the tubal peristalsis that could Preoperative diagnosis by transvaginal increase incidence of both tubal and ovarian USG and βHCG should be attempted but it is pregnancies (5). Five out of fourteen patients possible only in patients who present early had history of CuT use in our series. Recent with pelvic pain and vaginal bleeding. Most of literature quote increased incidence with the patients present with ruptured ectopic and infertility and assisted reproductive techniques are in circulatory collapse so preoperative (7, 8). diagnosis of ovarian ectopic on sonography is Two patients in our series had history of not easily made. Ovarian tumour producing primary infertility and one underwent HCG also may mislead the accurate intrauterine insemination in preceding cycle diagnosis. Intraoperative diagnosis could be and five patients had previous history of made in only 28% in a series of 25 cases, induced abortions. Choi et al reported because it was difficult to distinguish an and previous abdominal ovarian pregnancy from a hemorrhagic corpus surgeries are the most common risk factors in luteal cyst (2). his series of 49 cases of ovarian pregnancy Preoperative diagnosis was possible only but none of our patients had features of in two cases in our series. All fourteen endometriosis or history of abdominal surgery patients were confirmed by histopathology (11). Recently ovarian ectopic pregnancy has examination in our series. Ovarian pregnancy been reported after tubal ligation (12). had been treated by ipsilateral oophorectomy Although the ovary can accommodate itself in past, but the trend has been shifted toward more readily than the tube to the expanding conservative surgery such as cystectomy or pregnancy but 75% of ovarian gestation wedge resection performed at either

828 Iranian Journal of Reproductive Medicine Vol. 12. No. 12. pp: 825-830, December 2014 Ovarian pregnancy laparotomy or . Management References options like medical therapy with 1. Raziel A, Golan A, Pansky M, Ron-El R, Bukovsky I, , laparoscopic assisted medical Caspi E. Ovarian pregnancy: A report of twenty therapy with etoposide have been tried with cases in one institution. Am J Obstet Gynecol 1990; various success rate (14-16). 163: 1182-1185. Treatment of choice is resection of sac and 2. Hallatt JG. Primary ovarian pregnancy: a report of hemostasis preferably with laparoscopy or twenty-five cases. Am J Obstet Gynecol 1982; 143: 55-60. laparotomy as most of the patients arrive in 3. Studzinski Z, Branicka D, Filipczak A, Olinski K. collapsed state with uncertain diagnosis and Prolonged ovarian pregnancy: A case report. Ginekol laparoscopy may not be feasible (17). In our Pol 1999; 70: 33-35. (In Polish) series sac excision and hemostasis were 4. Sergent F, Mauger-Tinlot F, Gravier A, Verspyck E, performed in eleven cases as sacs were Marpean L. Ovarian pregnancies: revaluation of already ruptured with products of conception diagnostic criteria. Gynecol Obstet Biol Reprod (Paris) 2002; 31: 741-746. (In French) extruding in these patients. In two patients 5. Reichman J, Goldman JA, Feldberg D. Ovarian oopherectomy had to be resorted because of Pregnancy: Association with IUD, Pelvic pathology uncontrolled hemorrhage and hemostasis was and recurrent abortion. Eur J Obstet Gynecol Reprod not possible. Biol 1981; 12: 333-337. In case of term ovarian pregnancy excision 6. Joseph RJ, Irvine LM. Ovarian ectopic pregnancy: of placenta containing sac was performed. Aetiology, diagnosis, and challenges insurgical management. J Obstet Gynaecol 2012; 32: 472-474. Although none of the patients became 7. Dursun P, Gultekin M, Zeyneloglu HB. Ovarian pregnant in one year follow up in our series ectopic pregnancy after ICSI-ET: a case report and but literature suggest there is a high rate of literature review. Arch Gynecol Obstet 2008; 278: successful subsequent pregnancy and a low 191-193. rate of subsequent ectopic pregnancy or of 8. Comstock C, Huston K, Lee W. The ultrasonographic infertility (18). appearance of ovarian ectopic pregnancies. Obstet Gynecol 2005; 105: 42-45. 9. Chang FW, Chen CH, Liu JY. Early diagnosis of Conclusion ovarian pregnancy by ultrasound. Int J Gynecol Obstet 2004; 85: 186-187. Incidence of ovarian pregnancy is on rise 10. Sehgal A, Goyal LD, Goel P, Sunita BS. Full term due to increased incidence of infertility and ovarian pregnancy: A case report. Aust NZ J Obstet Gynecol 2005: 45: 165-166. use of assisted reproductive techniques. 11. Choi HJ, Im KS, Jung HJ, Lim KT, Mok JE, Kwon YS. Ultrasonography can detect ovarian Clinical analysis of ovarian pregnancy: a report of 49 gestations in unruptured cases but cannot cases. Eur J Obstet Gynecol Reprod Biol 2011; 158: easily differentiate ovarian from other tubal 87-89. gestation in ruptured state. As most of the 12. Vahnu C, Rajlaxmi M, Vandana R. Primary Ovarian patients present with ruptured sac in collapsed Pregnancy after Interval Tubal Ligation: A Case Report. J Fam Reprod Health 2013; 7: 187-188. state medical management is usually not 13. Ghi T, Banfi A, Marconi R, Iaco PD, Pilu G, Aloysio feasible. Conservative surgical approach DD, et al. Three-dimensional sonographic diagnosis remains the management of choice. of ovarian pregnancy. Ultrasound Obstet Gynecol 2005; 26: 102-104. Acknowledgments 14. Juan YC, Wang PH, Chen CH, Ma PC, Liu WM Successful treatment of ovarian pregnancy with laparoscopy-assisted local injection of etoposide. No financial support was taken for this Fertil Steril 2008; 90: 1200. study. 15. Mittal S, Dadhwal V, Baurasi P. Successful medical management of ovarian Mittal S, Dadhwal V, Baurasi P. Successful medical management of ovarian Conflict of interest pregnancy. Int J Gynecol Obstet 2003; 80: 309-310. 16. Bagga R, Suri V, Verma P, Chopra S, Kalra J. Failed The authors declare that they have no conflict Medical Management in Ovarian Pregnancy Despite of interests . Favorable Prognostic Factors-A Case Report. Med

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Gen Med 2006; 8: 35. 354-359. 17. Odejinmi F, Rizzuto MI, Macrae R, Olowu O, Hussain 18. Koo YJ, Choi HJ, Im KS, Jung HJ, Kwon YS M. Diagnosis and laparoscopic management of 12 Pregnancy outcomes after surgical treatment of consecutive cases of ovarian pregnancy and review ovarian pregnancy. Int J Gynaecol Obstet 2011; 114: of literature. J Minim Invasive Gynecol 2009; 16: 97-100.

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