Case Reports in Obstetrics and Gynecology

Case Reports in Obstetrics and Gynecology / 2016 / Article

Case Report | Open Access

Volume 2016 |Article ID 7618631 | https://doi.org/10.1155/2016/7618631

Yasushi Yamada, Satoshi Ohira, Teruyuki Yamazaki, Tanri Shiozawa, "Ectopic Molar Pregnancy: Diagnostic Efficacy of Magnetic Resonance Imaging and Review of the Literature", Case Reports in Obstetrics and Gynecology, vol. 2016, Article ID 7618631, 7 pages, 2016. https://doi.org/10.1155/2016/7618631

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

Academic Editor: Yoshio Yoshida
Received19 Jul 2016
Accepted07 Aug 2016
Published25 Aug 2016

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 [227] (Table 1).


Case numberAuthorAge (years)Gestation (weeks)SiteSymptomhCG typehCG level (mIU/mL)RupturePreoperative diagnosisTreatment

1Asseryanis et al. [2] (1993)2716Left tubePelvic massβ hCG3.5Ectopic molar pregnancyLeft tubal resection
2D’Aguillo et al. [3] (1982)2415Right ovaryAbdominal painβ hCG44000Right tubal pregnancyRSO
3Chase et al. [4] (1987)388Right tubeAmenorrheaβ hCG83Ectopic pregnancyLaparoscopic right tubal resection
4Chapman [5] (2001)359CervixVaginal bleedingβ hCG90181Cervical pregnancyD&C + laparoscopy
5Chauhan et al. [6] (2004)276Right tubeAbdominal painβ hCG406Right tubal pregnancyLaparoscopic right tubal resection
6Wu et al. [7] (2006)317Cesarean scarAbdominal pain Vaginal bleedingβ hCG61798Missed abortionD&C × 2
7Chauhan et al. [8] (2006)4012Left cornuaAbdominal pain Vaginal bleedingβ hCG2905UnknownTAH
8Tulon et al. [9] (2010)307Left tubeAbdominal pain Vaginal bleedingβ hCG5308+Ruptured ectopic pregnancyLeft tubal resection
9Hwang et al. [10] (2010)4112Left cornuaVaginal bleedingβ hCG57738Ectopic pregnancyLaparoscopic left cornual resection
10Juan [11] (2013)208Left tubeAbdominal painβ hCG6984+Left tubal pregnancyLaparoscopic left tubal resection
11Mbarki et al. [12] (2015) 326Left tubeAbdominal pain Vaginal bleedingβ hCG404000+Left tubal pregnancyLaparoscopic left tubal resection
12377Left tubeAbdominal pain Shock vitalβ hCG290600+Ruptured ectopic pregnancyLaparoscopic left tubal resection
13Jock et al. [13] (1981)2712Left ovaryAmenorrheaSerum hCG165000+Ovarian choriocarcinomaLSO + OM + D&C
14Zite et al. [14] (2002)Unknown12Right cornuaAbdominal painSerum hCG97000+Intrauterine mole + ovarian bleedingRight cornual resection + D&C
15Mohamed and Sharma [15] (2003)32UnknownRight tubeAbdominal pain Vaginal bleedingSerum hCG7823+Ectopic pregnancyRight tubal resection
16Church et al. [16] (2008)296Left ovaryAbdominal pain Vaginal bleedingSerum hCG3584+Left tubal pregnancyLSO
17Leung et al. [17] (2010)38UnknownUterus + right ovaryVaginal bleedingSerum hCG54000+Intrauterine moleDC → right ovarian resection
18Bousfiha et al. [18] (2012)326Left tubeAbdominal pain Vaginal bleedingSerum hCG3454Ectopic pregnancyLaparoscopic left tubal resection
19Sehn et al. [19] (2013)20UnknownLeft ovaryAbdominal pain Vaginal bleedingSerum hCG100355+UnknownLaparoscopic left ovarian resection → laparoscopic LSO
20Ota et al. [20] (2014)238PeritoneumAbdominal pain Shock vitalSerum hCG8000+Ruptured ectopic pregnancyLaparotomy
21Ikuma et al. [21] (1992)4411Left tubeVaginal bleedingUrine hCG1600+GTDMTX 1corse → ATH + LSO → MTX 1corse
22P. Dumitrescu and A. Dumitrescu [22] (1960)28UnknownPeritoneumUnknownNot performed+Ectopic pregnancyLaparotomy
23Westerhout Jr. [23] (1964)328–10Left tubeAbdominal pain Vaginal bleeding Shock vitalNot performed+UnknownLSO
24Pour-Reza [24] (1974)36UnknownLeft tubeAbdominal pain Vaginal bleedingNot performed+Ectopic pregnancyLSO
25Farrukh et al. [25] (2007)27UnknownRight tubeAbdominal pain Vaginal bleedingNot performed+Ectopic pregnancyLaparoscopic right tubal resection
26Samaila et al. [26] (2009) 20UnknownTubeAbdominal pain Vaginal bleedingNot performed+UnknownLaparoscopic tubal resection
2728UnknownTubeAbdominal pain Vaginal bleedingNot performed+UnknownLaparoscopic tubal resection
2833UnknownTubeAbdominal pain Vaginal bleedingNot performed+UnknownLaparoscopic tubal resection
2935UnknownTubeAbdominal pain Vaginal bleedingNot performed+UnknownLaparoscopic tubal resection
3037UnknownTubeAmenorrheaNot performed+UnknownLaparoscopic tubal resection + LM
31Yakasai et al. [27] (2012)3512Left tubeAbdominal painNot performed+Left tubal pregnancyLeft tubal resection
32Current case338Right cornuaAmenorrheaβ hCG66400 (ng/mL)Ectopic molar pregnancyTAH

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).

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.

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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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.


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