Case Reports in Obstetrics and Gynecology
Volume 2016 (2016), Article ID 7618631, 7 pages
http://dx.doi.org/10.1155/2016/7618631

Case Report

Ectopic Molar Pregnancy: Diagnostic Efficacy of Magnetic Resonance Imaging and Review of the Literature

Yasushi Yamada,¹ Satoshi Ohira,¹ Teruyuki Yamazaki,² and Tanri Shiozawa¹

¹Department of Obstetrics and Gynecology, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
²Department of Obstetrics and Gynecology, Iida Municipal Hospital, 438 Yawatamachi, Iida 395-8502, Japan

Received 19 July 2016; Accepted 7 August 2016

Academic Editor: Yoshio Yoshida

Copyright © 2016 Yasushi Yamada et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract.

Ectopic molar pregnancy is extremely rare, and preoperative diagnosis is difficult. Our literature search found only one report of molar pregnancy diagnosed preoperatively. Moreover, there is no English literature depicting magnetic resonance image (MRI) findings of ectopic molar pregnancy. We report a case of ectopic molar pregnancy preoperatively diagnosed using MRI. A literature review of 31 cases of ectopic molar pregnancy demonstrated that lesions have been found in the fallopian tube (19 cases, 61%), ovary (5 cases, 16%), cornu (3 cases, 10%), peritoneum (2 cases, 6%), uterine cervix (1 case, 3%), and cesarean scar (1 case, 3%). Abdominal pain and abnormal vaginal bleeding were reported in 70% and 61% of the patients, respectively. Twenty-one cases (67%) presented with rupture and hemoperitoneum. All patients underwent surgical resection or dilatation and curettage. Methotrexate therapy was performed in one case because residual trophoblastic tissue was suspected. A second operation was performed in one case of ovarian molar pregnancy because serum hCG levels increased again after primary focal ovarian resection. No patients developed metastatic disease or relapsed. These findings suggest the prognosis of ectopic molar pregnancy to be favorable.

1. Introduction

Gestational trophoblastic disease (GTD) consists of hydatidiform mole, choriocarcinoma, placental site trophoblastic tumor, and epithelioid trophoblastic tumor. Because the majority of GTD cases occur in the uterus, ectopic molar pregnancy is extremely rare. Gillespie et al. estimated that the incidence of ectopic GTD is 1.5 per one million births in the UK [1]. Preoperative diagnosis of ectopic molar pregnancy is difficult, and our literature search found only one report of molar pregnancy diagnosed preoperatively [2]. Moreover, there is no English literature depicting magnetic resonance image (MRI) findings of ectopic molar pregnancy. Here, we report the first case of ectopic molar pregnancy preoperatively diagnosed using MRI, with a review of the literature.

2. Literature

We performed a review of all ectopic molar pregnancy cases published in English and Japanese between 1960 and 2014. All studies were obtained from Medline using the terms “ectopic molar pregnancy”, and from references of the articles. All articles without an abstract or with unavailable full text were excluded. We identified 26 articles reporting 31 cases of ectopic molar pregnancy [2–27] (Table 1).

Table 1: Thirty-one cases and the current case on ectopic molar pregnancy.

Case number
Author
Age (years)
Gestation (weeks)
Site
Symptom
hCG type
hCG level (mIU/mL)
Rupture
Preoperative diagnosis
Treatment

1
Asseryanis et al. [2] (1993)
27
16
Left tube
Pelvic mass
β hCG
3.5
−
Ectopic molar pregnancy
Left tubal resection

2
D’Aguillo et al. [3] (1982)
24
15
Right ovary
Abdominal pain
β hCG
44000
−
Right tubal pregnancy
RSO

3
Chase et al. [4] (1987)
38
8
Right tube
Amenorrhea
β hCG
83
−
Ectopic pregnancy
Laparoscopic right tubal resection

4
Chapman [5] (2001)
35
9
Cervix
Vaginal bleeding
β hCG
90181
−
Cervical pregnancy
D&C + laparoscopy

5
Chauhan et al. [6] (2004)
27
6
Right tube
Abdominal pain
β hCG
406
−
Right tubal pregnancy
Laparoscopic right tubal resection

6
Wu et al. [7] (2006)
31
7
Cesarean scar
Abdominal pain Vaginal bleeding
β hCG
61798
−
Missed abortion
D&C × 2

7
Chauhan et al. [8] (2006)
40
12
Left cornua
Abdominal pain Vaginal bleeding
β hCG
2905
−
Unknown
TAH

8
Tulon et al. [9] (2010)
30
7
Left tube
Abdominal pain Vaginal bleeding
β hCG
5308
+
Ruptured ectopic pregnancy
Left tubal resection

9
Hwang et al. [10] (2010)
41
12
Left cornua
Vaginal bleeding
β hCG
57738
−
Ectopic pregnancy
Laparoscopic left cornual resection

10
Juan [11] (2013)
20
8
Left tube
Abdominal pain
β hCG
6984
+
Left tubal pregnancy
Laparoscopic left tubal resection

11
Mbarki et al. [12] (2015)
32
6
Left tube
Abdominal pain Vaginal bleeding
β hCG
404000
+
Left tubal pregnancy
Laparoscopic left tubal resection

12
37
7
Left tube
Abdominal pain Shock vital
β hCG
290600
+
Ruptured ectopic pregnancy
Laparoscopic left tubal resection

13
Jock et al. [13] (1981)
27
12
Left ovary
Amenorrhea
Serum hCG
165000
+
Ovarian choriocarcinoma
LSO + OM + D&C

14
Zite et al. [14] (2002)
Unknown
12
Right cornua
Abdominal pain
Serum hCG
97000
+
Intrauterine mole + ovarian bleeding
Right cornual resection + D&C

15
Mohamed and Sharma [15] (2003)
32
Unknown
Right tube
Abdominal pain Vaginal bleeding
Serum hCG
7823
+
Ectopic pregnancy
Right tubal resection

16
Church et al. [16] (2008)
29
6
Left ovary
Abdominal pain Vaginal bleeding
Serum hCG
3584
+
Left tubal pregnancy
LSO

17
Leung et al. [17] (2010)
38
Unknown
Uterus + right ovary
Vaginal bleeding
Serum hCG
54000
+
Intrauterine mole
D＆C → right ovarian resection

18
Bousfiha et al. [18] (2012)
32
6
Left tube
Abdominal pain Vaginal bleeding
Serum hCG
3454
−
Ectopic pregnancy
Laparoscopic left tubal resection

19
Sehn et al. [19] (2013)
20
Unknown
Left ovary
Abdominal pain Vaginal bleeding
Serum hCG
100355
+
Unknown
Laparoscopic left ovarian resection → laparoscopic LSO

20
Ota et al. [20] (2014)
23
8
Peritoneum
Abdominal pain Shock vital
Serum hCG
8000
+
Ruptured ectopic pregnancy
Laparotomy

21
Ikuma et al. [21] (1992)
44
11
Left tube
Vaginal bleeding
Urine hCG
1600
+
GTD
MTX 1corse → ATH + LSO → MTX 1corse

22
P. Dumitrescu and A. Dumitrescu [22] (1960)
28
Unknown
Peritoneum
Unknown
Not performed
—
+
Ectopic pregnancy
Laparotomy

23
Westerhout Jr. [23] (1964)
32
8–10
Left tube
Abdominal pain Vaginal bleeding Shock vital
Not performed
—
+
Unknown
LSO

24
Pour-Reza [24] (1974)
36
Unknown
Left tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Ectopic pregnancy
LSO

25
Farrukh et al. [25] (2007)
27
Unknown
Right tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Ectopic pregnancy
Laparoscopic right tubal resection

26
Samaila et al. [26] (2009)
20
Unknown
Tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Unknown
Laparoscopic tubal resection

27
28
Unknown
Tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Unknown
Laparoscopic tubal resection

28
33
Unknown
Tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Unknown
Laparoscopic tubal resection

29
35
Unknown
Tube
Abdominal pain Vaginal bleeding
Not performed
—
+
Unknown
Laparoscopic tubal resection

30
37
Unknown
Tube
Amenorrhea
Not performed
—
+
Unknown
Laparoscopic tubal resection + LM

31
Yakasai et al. [27] (2012)
35
12
Left tube
Abdominal pain
Not performed
—
+
Left tubal pregnancy
Left tubal resection

32
Current case
33
8
Right cornua
Amenorrhea
β hCG
66400 (ng/mL)
−
Ectopic molar pregnancy
TAH

hCG: human chorionic gonadotropin; TAH: total abdominal hysterectomy; LSO: left salpingo-oophorectomy; RSO: right salpingo-oophorectomy; D&C: dilatation and curettage; OM: omentectomy; LM: laparoscopic myomectomy; MTX: methotrexate; GTD: gestational trophoblastic disease.

3. Clinical Case

We recently observed a 33-year-old, gravida 3 para 2, woman who visited our hospital with a complaint of amenorrhea for 8 weeks and 3 days since her last menstrual period. Her blood pressure was 104/76 mmHg, with pulse of 68 beats per minute. Her abdomen was soft and she had no tenderness on palpation. On vaginal examination, the uterus was asymmetrically enlarged. Transvaginal ultrasonography (TVUS) revealed an empty endometrial cavity and right cornual hyperechoic mass (5 cm) with multiple vesicles (Figure 1). Serum beta human chorionic gonadotropin (β-hCG) level was 66,400 ng/mL. Because molar ectopic pregnancy was suspected and her vital signs were stable, MRI was performed. MRI revealed a 5 cm mass on the right cornu, of isosignal intensity on T1-weighted images (T1-WI) and high signal intensity on T2-weighted images (T2-WI). The mass included vesicles with low signal intensities on T1-WI and high signal intensities on T2-WI, suggesting hydropic villi. The mass showed strong gadolinium contrast enhancement, and its margins were clear. Several flow voids were observed at the edge of the mass (Figure 2).

Figure 1: Transvaginal ultrasonographic image at 8 weeks of gestation. A hyperechoic mass (5 cm) in the right cornu containing multiple vesicles (arrows).

(a)

(b)

(c)

Figure 2: Coronal MRI images. (a) T1-WI shows an isosignal intensity mass in the right cornu. The mass includes a cyst (arrow) and shows low signal intensity. Several flow voids (arrow heads) are observed at the edge of the mass. (b) T2-WI. The mass and cyst (arrow) show high signal intensity. Several flow voids (arrow heads) are observed at the edge of the mass. (c) Gadolinium-enhanced and fat-suppressed T1-WI demonstrates a well-enhanced mass and clear margins. Arrow indicates the cyst.

According to these findings, a preoperative diagnosis of ectopic molar pregnancy in the right uterine cornu was made. Because the patient no longer had any wish for a baby, an abdominal hysterectomy was performed. We chose not a laparoscopic surgery but a laparotomy to avoid rupture of enlarged uterine cornu during removing of the uterus through the vagina. On laparotomy, a dark-blue mass with increased vascularity in the right uterine cornu was noted (Figure 3(a)). Both adnexa were normal, and there was no hemoperitoneum. Total abdominal hysterectomy was performed because the patient and her husband did not wish to preserve fertility. Grossly, cut sections of the uterus showed a dark-red 4 cm mass with small vesicles in the right cornu. The uterus had no malformation such as unicornuate or bicornuate uterus. On pathology, chorionic villi with focal trophoblastic proliferation and hydropic change were observed. There was no cistern formation. A few proliferating stromal cells were observed but degeneration was not noted (Figure 3(b)). Invasion of trophoblasts to the myometrium was noted (Figure 3(c)). The postoperative diagnosis was ectopic invasive mole in the right cornu. Systemic computed tomography was performed after operation and revealed no metastatic lesion. The patient was followed up weekly or biweekly, and her β-hCG level was negative 8 weeks postoperatively. The patient has been free from relapse for 60 months.

(a)

(b)

(c)

Figure 3: (a) Photograph of the uterus during laparotomy. A dark-blue mass is seen in the right cornu, with increased vascularity (arrow). (b) Enlarged hydropic villi and focal trophoblastic proliferation (asterisk) are observed. There is no cistern formation. A few proliferating stromal cells are observed but degeneration is not noted. (c) Invasion of villi and trophoblasts to the uterine myometrium. The trophoblastic proliferation is variable.

4. Results (Table 1)

Of the 31 cases reviewed, the mean age was 31.3 years (20 to 44 years), and the lesions were found in the fallopian tube (19 cases, 61%), ovary (5 cases, 16%), cornu (3 cases, 10%), peritoneum (2 cases, 6%), uterine cervix (1 case, 3%), and cesarean scar (1 case, 3%). Abdominal pain and abnormal vaginal bleeding were reported in 70% and 61% of the patients, respectively. Twenty-one cases (67%) presented with rupture and hemoperitoneum. Serum β-hCG levels in 12 cases and serum hCG levels in 8 patients ranged within 3.5–404,000 mIU/mL and 3,454–165,000 mIU/mL, respectively. All patients underwent operation or dilatation and curettage. A second operation was needed in one ovarian molar pregnancy case because serum hCG levels increased again after primary focal ovarian resection. Methotrexate therapy was performed in one case because residual trophoblastic tissue was suspected. None of the patients developed metastatic disease or relapsed.

5. Discussion

Preoperative diagnosis of ectopic molar pregnancy is difficult, and we found only one reported case. Asseryanis et al. preoperatively detected a left tubal molar pregnancy using transvaginal color-flow Doppler, revealing an arteriovenous shunt flow of both the tumor and myometrium [2]. However, the efficacy of transvaginal color-flow Doppler in the diagnosis of ectopic molar pregnancy remains controversial [28]. We suspected cornual molar pregnancy because transvaginal ultrasonography revealed a mass with small vesicles in the right cornu, which is a typical finding of molar pregnancy. MRI revealed a right cornual mass with isosignal intensity on T1-weighted images (T1-WI) and high signal intensity on T2-weighted images (T2-WI). In addition, small vesicles in the mass showed low signal intensities on T1-WI and high signal intensities on T2-WI, which suggested hydropic villi. Distinguishing between ectopic molar pregnancy and choriocarcinoma or “ordinary” ectopic pregnancy is important. Ha et al. reported four important MRI findings for the differential diagnosis between uterine choriocarcinoma and uterine invasive mole: (i) the tumor margin is well-defined in choriocarcinoma and ill-defined in invasive mole; (ii) the hyperintensity pattern on T1-WI is nodular in choriocarcinoma and scattered in invasive mole; (iii) intratumoral vascularity is absent or minimal in choriocarcinoma due to severe central necrosis and hemorrhage, whereas intratumoral vascularity is increased and the tumor is densely enhanced in invasive mole; and (iv) invasive mole has molar tissue-like tiny cystic lesions within the mass [29]. Although the disease site differed, their suggestions may be useful for diagnosing ectopic molar pregnancy. In our case, molar tissue-like tiny cystic lesions, intratumoral hypervascularity, and dense enhancement were observed. We believe that MRI is a powerful tool for diagnosis of ectopic molar pregnancy. However, there may have been several cases in which MRI was not performed due to shock induced by rupture.

The rate of rupture and hemoperitoneum in cases of molar ectopic pregnancy rupture was 67%. Berlingieri et al. and Frates et al. reported rates of ruptured normal ectopic pregnancy of 29.5% and 25.2%, respectively [30, 31], demonstrating that the rate of molar ectopic pregnancy rupture was significantly higher than that of normal ectopic pregnancy. This may be due to the higher invasive ability of trophoblasts in gestational trophoblastic disease compared with trophoblasts in normal pregnancy.

The serum β-hCG levels in 12 cases and serum hCG levels of 8 patients ranged within 3.5–404,000 mIU/mL and 3,454–165,000 mIU/mL, respectively. Frates et al. reported that the serum hCG levels of 225 normal ectopic pregnancy ranged within 7–107,949 mIU/mL [31]. Tasha et al. reported 18 cases of ectopic gestational trophoblastic disease in 100 cases of ectopic pregnancy. The hCG levels of normal ectopic pregnancy were 1,256–13,494 mIU/mL, partial mole 6,642–15,678 mIU/mL, and complete mole 7,920–24,733 mIU/mL. Furthermore, cases of intrauterine molar pregnancy are known to have higher hCG levels than normal pregnancies. Although Chauhan et al. suggested that implantation in the fallopian tube might preclude adequate vascularization and lead to low hCG levels in ectopic molar pregnancy [6], these reports suggested that ectopic molar pregnancy cannot be distinguished from normal ectopic pregnancy by hCG levels alone. Because none of the patients developed metastatic disease or relapsed, the prognosis of molar ectopic pregnancy is suggested to be favorable.

Competing Interests

The authors declare that they have no competing interests.

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