GENERAL GYNECOLOGY

Simultaneous presentation of tubal and primary abdominalpregnancies following clomiphene citrate treatment

Tsuyoshi Baba • Toshiaki Endo • Keiko Ikeda •

Naoko Takenami • Ayumi Shimizu • Miyuki Morishita •

Hiroyuki Honnma • Hideyuki Ikeda • Tsuyoshi Saito

Received: 1 February 2012 / Accepted: 14 March 2012 / Published online: 28 March 2012

� Springer-Verlag 2012

Abstract Abdominal pregnancy is a rare condition that is

potentially life-threatening for the mother. We present a

case of simultaneous ectopic pregnancies (EPs) in the right

fallopian tube and in the vesicouterine pouch. A 26-year-

old woman had undergone prior ovulation induction with

clomiphene citrate and human chorionic gonadotropin

(hCG) at an outside hospital for unexplained infertility. The

patient was referred to our hospital for a suspected ectopic

pregnancy at 6 weeks gestation. Transvaginal ultrasonog-

raphy detected a viable fetus at the anterior left side of the

uterus; therefore, we suspected a left tubal pregnancy.

However, laparoscopic surgery revealed that EPs were

located in both the left vesicouterine pouch and in the right

fallopian tube. Resection of the right salpinx and abdomi-

nal implant were performed. Histopathological examina-

tion confirmed the simultaneous presentation of a primary

abdominal pregnancy and a right tubal pregnancy. After

surgery, the patient’s serum hCG level returned to normal.

Concurrent EPs and abdominal pregnancy are very rare.

However, it should be noted that reproductive technologies

sometimes cause unusual clinical situations. A thorough

abdominal inspection is needed.

Keywords Abdominal pregnancy � Clomiphene citrate �Ectopic pregnancy � Laparoscopic surgery

Introduction

An ectopic pregnancy (EP) is an implantation of the blas-

tocyst outside the uterine cavity. The overall incidence of

EP increased during the mid-twentieth century, plateauing

at approximately 2 % in the early 1990s [1]. It is difficult to

determine the current incidence of EP because inpatient

hospital treatment of EPs has decreased and data from

hospitalization records are not available. Widespread use of

reproductive techniques, such as cleavage-stage embryo

transfer and ovulation induction, is thought to elevate the

risk of EP. The prevalence of EP has been increased to

2–11 % by assisted reproductive technology (ART) [2, 3].

In patients undergoing ovulation induction, the reported

incidence of EP is approximately 3 % [4]. The location of

EPs vary, but the vast majority ([95 %) are found in the

fallopian tubes. In spite of the increased incidence of EP

after ovulation induction and ART, abdominal pregnancy is

still a rare phenomenon. Therefore, a patient with two

simultaneous primary EPs in different locations, the

abdominal cavity and the fallopian tube, is an extremely

rare case. The present case involves EPs that developed

during an ovulation induction treatment cycle.

Case report

A 26-year-old Japanese woman, gravida 0, para 0, had

visited an outside hospital with sustained high basal body

temperature. Her past medical history was nothing. She had

undergone her first cycle of controlled ovarian stimulation

using 100 mg/day of clomiphene citrate beginning on cycle

day 5 and lasting for 5 days. Urinary LH monitoring,

determined daily with the use of commercial kits, was used

to time intercourse. Pregnancy was confirmed, and it was

T. Baba (&) � T. Endo � K. Ikeda � N. Takenami �A. Shimizu � M. Morishita � H. Honnma � T. Saito

Department of Obstetrics and Gynecology,

Sapporo Medical University, South 1 West 16, Sapporo,

Hokkaido 060-8543, Japan

e-mail: tbaba@sapmed.ac.jp

H. Ikeda

Department of Pathology, Rumoi City Hospital,

2-16-1 Shinonome, Rumoi, Hokkaido 077-8511, Japan

123

Arch Gynecol Obstet (2012) 286:395–398

DOI 10.1007/s00404-012-2300-z

estimated to be 4 weeks gestation from the day of ovula-

tion. Two weeks later, she visited the hospital again with

intermittent abdominal pain in her left lower quadrant and

2 days of vaginal bleeding. An ultrasonographic scan did

not detect a gestational sac in the uterine cavity. She was

referred to our hospital for a suspected EP. A transvaginal

ultrasound examination showed a small gestational sac

surrounded by a hematoma and a fetal heartbeat located in

the anterior left side of the uterus. Bimanual examination

revealed tenderness in the same location. Her serum hCG

levels were elevated at 5,349.2 mIU/ml. Based on these

findings, she was initially diagnosed with a left tubal

pregnancy. Laparoscopic surgery was performed and

showed a small amount of hemoperitoneum and an

enlarged but unruptured right fallopian tube (Fig. 1). The

left fallopian tube was normal, in contrast to our expecta-

tions, and no adhesions were observed in the pelvis. A right

salpingectomy and hemostasis was performed with mono-

polar forceps. A thorough abdominal inspection revealed

that a small (approximately 1.0–1.5 cm in diameter)

hematoma was buried in the vesicouterine pouch adjacent

to the left round ligament (contralateral to the hemosal-

pinx) (Fig. 2), and the mass was completely removed.

Histopathological examination confirmed that chorionic

villi were seen in both the right salpinx and the hematoma

(Fig. 3). The patient had an uneventful postoperative

course, and was discharged 5 days after surgery.

Discussion

Timely diagnosis of EPs is essential because they may

cause life-threatening hemorrhage. Despite the recent

advances in diagnostic imaging apparatus, it is still difficult

to accurately diagnose the site of an EP. The sensitivity of

transvaginal ultrasound for the diagnosis of EP ranges

69–93 % and is affected by the gestational age, the exis-

tence of adnexal masses, and the diagnostic ability of the

physician [5–7]. It is not surprising that misdiagnoses occur

in cases of absent adnexal masses, such as abdominal

pregnancy. Abdominal pregnancy is a rare phenomenon

that occurs in approximately 1 in 10,000 pregnancies [8]. It

is a serious disease because it is seldom detected until an

advanced gestational age or in the event of subsequent

severe hemorrhage [9]. Maternal mortality rates associated

with abdominal pregnancy have been reported to be in the

range 5–12 % [10, 11]. In other report, maternal mortality

rate of abdominal pregnancy is estimated to be 7–8 times as

high as that of other types of ectopic pregnancy [12, 13].

Fortunately, in the present case, because of the existence of

conceptus in the pelvis and the thorough abdominal

inspection, we detected the abdominal EP at an early stage

and successfully treated the patient.

There are no specific ultrasonographic characteristics of

abdominal pregnancies at an early gestational age. The

classic ultrasound findings, which can be observed at an

advanced gestational age, are the absence of the uterine

wall between the bladder and the fetus, poor visualization

of the placenta, extrauterine location of the placenta, and a

fetus located adjacent to the mother’s abdominal organs

[14]. Therefore, magnetic resonance imaging (MRI) and

laparoscopy should be considered in the cases of suspected

abdominal pregnancy.

The etiology of abdominal pregnancy is thought to

involve two mechanisms: (1) direct implantation on the

peritoneum (primary abdominal pregnancy) and (2) abor-

tion or rupture of a tubal (less commonly, an ovarian)

pregnancy and subsequent re-implantation of the conceptus

on the peritoneum (secondary abdominal pregnancy). Most

of the abdominal pregnancies are considered secondary.

The implantation sites of abdominal pregnancies are

mostly the pouch of Douglas and the posterior uterine wall,

maybe because adnexa are usually located in these sites

[15]. Based on the literature, the following is the suggested

diagnostic criteria for primary abdominal pregnancy: (1)

intact ovary and fallopian tube, (2) absence of uteroperi-

toneal fistula, (3) existence of only peritoneal implantation,

and (4) early gestational age (which eliminates the possi-

bility of secondary implantation) [16]. Our case had both

tubal and peritoneal lesions, which did not fulfill the cri-

teria of primary abdominal pregnancy. However, repro-

ductive technologies such as ovulation induction and ART,

which tend to cause multiple pregnancies, are unexpected

circumstances in the previous report. This case involved a

pregnancy at an early gestational age, making secondary

implantation unlikely. Therefore, we think that this case

had two distinct pregnancies: a right tubal pregnancy and a

primary abdominal pregnancy.Fig. 1 Laparoscopic findings of the tubes. Right hemosalpinx

(arrow) and intact left fallopian tube are shown

396 Arch Gynecol Obstet (2012) 286:395–398

123

In conclusion, a high index of suspicion is important for

diagnosing abdominal pregnancy and reducing the associ-

ated morbidity and mortality. Multiple implantation sites

should be considered, particularly in the cases with mul-

tiple ovulations or embryo transfers.

Acknowledgments We appreciate Dr. Hiroyasu Konno for his

helpful suggestion.

Conflicts of interest None.

References

1. Centers for Disease Control and Prevention (CDC) (1995)

Ectopic pregnancy—United States, 1990–1992. Morb Mortal

Wkly Rep 44:46–48

2. Nazari A, Askari HA, Check JH, O’Shaughnessy A (1993)

Embryo transfer technique as a cause of ectopic pregnancy in in

vitro fertilization. Fertil Steril 60:919–921

3. Clayton HB, Schieve LA, Peterson HB, Jamieson DJ, Reynolds

MA, Wright VC (2006) Ectopic pregnancy risk with assisted

reproductive technology procedures. Obstet Gynecol

107:595–604

4. McBain JC, Eevans JH, Pepperell RJ, Robinson HP, Smith MA,

Brown JB (1980) An unexpectedly high rate of ectopic pregnancy

following the induction of ovulation with human pituitary and

chorionic gonadotrophin. BJOG 87:5–9

5. Shalev E, Yarom I, Bustan M, Weiner E, Ben-Shlomo I (1998)

Transvaginal sonography as the ultimate diagnostic tool for the

management of ectopic pregnancy: experience with 840 cases.

Fertil Steril 69:62–65

6. Condous G, Lu C, Van Huffel SV, Timmerman D, Bourne T

(2004) Human chorionic gonadotrophin and progesterone levels

for the investigation of pregnancies of unknown location. Int J

Gynaecol Obstet 86:351–357

Fig. 2 Laparoscopic findings of the pelvic cavity. Slight hemoperitoneum and peritoneal implant (arrow) adjacent to the left round ligament are

shown (a low magnification, b high magnification)

Fig. 3 Histopathological findings of ectopic pregnancy (hematoxy-

lin–eosin stain, a vesicouterine pouch. Chorionic villi in hematoma.

Original magnification 940; b right fallopian tube. Chorionic villi

corresponding to first trimester surrounded by decidualized tubal

stroma. Original magnification 912.5)

Arch Gynecol Obstet (2012) 286:395–398 397

123

7. Kaplan BC, Dart RG, Moskos M, Kuligowska E, Chun B, Adel

Hamid M, Northern K, Schmidt J, Kharwadkar A (1996) Ectopic

pregnancy: prospective study with improved diagnostic accuracy.

Ann Emerg Med 28:10–17

8. Atrash HK, Friede A, Hogue CJ (1987) Abdominal pregnancy in

the United States: frequency and maternal mortality. Obstet

Gynecol 69:333–337

9. Shafi SM, Malla MA, Salaam PA, Kirmani OS (2009) Abdominal

pregnancy as a cause of hemoperitoneum. J Emerg Trauma Shock

2:196–198

10. Nkusu Nunyalulendho D, Einterz EM (2008) Advanced abdom-

inal pregnancy: case report and review of 163 cases reported

since 1946. Rural Remote Health 8:1087. Available from

http://www.rrh.org.au. Accessed on 23 May 2009

11. Sunday-Adeoye H, Twomey D, Egwuatu EV, Okonta PI (2011)

A 30-year review of advanced abdominal pregnancy at the Mater

Misericordiae Hospital, Afikpo, southeastern Nigeria

(1976–2006). Arch Gynecol Obstet 283:19–24

12. Hornemann A, Holl-Ulrich K, Finas D, Altgassen C, Diedrich K,

Hornung D (2008) Laparoscopic management of early primary

omental pregnancy. Fertil Steril 89:991.e9–991.e11

13. da Silva BB, de Araujo EP, Cronemberger JN, dos Santos AR,

Lopes-Costa PV (2008) Primary twin omental pregnancy: report of

a rare case and literature review. Fertil Steril 90:2006.e13–2006.e25

14. Varma R, Mascarenhas L, Jame D (2003) Successful outcome of

advanced abdominal pregnancy with exclusive omental insertion.

Ultrasound Obstet Gynecol 21:192–194

15. Goh TH, Rahman SA (1980) Primary peritoneal pregnancy

implanted on the uterine fundus. Aust N Z J Obstet Gynaecol

20:240–241

16. Studdiford WE (1942) Primary peritoneal pregnancy. Am J

Obstet Gynecol 44:487–491

398 Arch Gynecol Obstet (2012) 286:395–398

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