Gastrointestinal Bleeding Caused by Ileal Metastasis of a Tubal Complete Mole: A Case Report

Introduction

Hydatidiform mole is a type of pregnancy characterized by vesicular swelling of placental villi and usually the absence of an intact fetus. It is one of several correlated disorders of fertilization that are referred to as the gestational trophoblastic diseases (GTDs). GTD is a disease of the proliferative trophoblastic allograft and is histologically classified into 5 types; partial mole, complete hydatidiform mole, invasive and metastatic mole, choriocarcinoma, and placental site trophoblastic tumor. ^1,2 On clinically monitoring the serum human gonadotrophin hormones (hCG) in patients with a history of molar pregnancy, it was observed that 10%–20% of patients had rising or persistently raised hCG levels. This condition has been variously termed by different research groups as “gestational trophoblastic tumor,” “persistent GTD,” “residual GTD,” “malignant GTD,” and “gestational trophoblastic neoplasia (GTN).” At the Internation Federation of Gynecology and Obstetrics (FIGO) 2000 meeting, the term “GTN” was recommended to replace all other terminologies to avoid confusion. ³

Even though the known prevalence of GTD in ectopic pregnancy was 0.16 out of 1,000 deliveries, nearly 40 cases of ectopic tubal hydatidiform mole have been reported. ^4,5 There have been a few case reports of choriocarcinoma presenting gastrointestinal tract metastasis. ⁶ However, a tubal complete mole with concurrent ileal metastasis has never yet been reported. Here, we present a patient with a complete mole embedded in a tubal pregnancy and ileal metastatic focus.

Materials and Methods

A 33-year-old (gravida 3 and para 2) woman was referred to our clinic with unknown hemoperitoneum. She had undergone laparoscopic left salpingectomy for a tubal complete mole a month earlier. Her serum β-hCG was 20,353 IU/L at that time. Just before her referral, she underwent diagnostic laparoscopy in a local gynecologic clinic.

No causal factors for the hemoperitoneum were found in the abdominopelvic cavity. Upon admission, she complained of profuse gastrointestinal bleeding that had lasted for several days. She was followed with hCG measurements every week and there was a rise in hCG level for three weeks to 74,434 IU/L. The diagnosis for GTN was confirmed. Fiberscopic duodenogastroscopy and sigmoidoscopy showed no bleeding foci; however, an abdominopelvic computed tomography revealed a 1.5 cm-sized polypoid mass in the ileum as a suspicious bleeding focus. Mesenteric angiography showed a focal round and tubular lesion in the ileal branch.

Chest radiography and computed tomography showed no abnormalities. The initial tubal mole was histologically confirmed by a gynecologic pathologist as a complete hydatidiform mole. The patient underwent laparotomy, and an ileal invasion of gestational tissue was identified. Consequently small bowel resection and anastomosis were performed (Fig. 1). A histological examination confirmed the diagnosis of a complete hydatidiform mole (Figs. 2, 3).

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FIG. 1.

Photograph of an ileal specimen showing a 2 cm-sized polypoid hemorrhagic mass.

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FIG. 2.

A hydropic villus surrounded by marked trophoblastic proliferation and blood clots (× 100, H&E).

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FIG. 3.

Frequent mitoses and cytological atypism are found in proliferating syncytioblasts and cytotrophoblasts (× 200, H&E).

A FIGO stage of IV and a prognostic score of 2 were confirmed, based on the current FIGO rating system. ¹ Currently, the treatment regimen for low-risk group patients requires single methotrexate chemotherapy for optimal results. ^7,8 Thus, the patient received nine cycles of adjuvant methotrexate chemotherapy. After each cycle, a quantitative β-hCG titer was taken until three successive titers were negative. Patient counseling concerning the diagnosis, follow-up, and contraception was performed. Monitoring of the patient's β-hCG revealed maintenance of normal levels for 20 consecutive months.

Discussion

GTN includes invasive mole, choriocarcinoma, and placental site trophoblastic tumors. The overall cure rate in treating these tumors currently exceeds 90%. Thorough evaluation and staging allow selection of appropriate therapy that maximizes chances for cure while minimizing toxicity. Nonmetastatic (stage I) and low-risk metastatic (stages II and III, World Health Organization score < 7) GTN can be treated with single-agent chemotherapy, resulting in a survival rate approaching 100%. High-risk metastatic GTN (stage IV, WHO score > or = 7) requires initial multiagent chemotherapy with or without adjuvant radiation and surgery to achieve a survival rate of 80% to 90%. ⁹

Lower gastrointestinal bleeding is defined as an abnormal intraluminal blood loss from a source distal to the ligament of Treitz and accounts for approximately 20% of gastrointestinal bleeding. Lower gastrointestinal bleeding can be because of numerous conditions, including diverticulosis, anorectal diseases, benign or malignant neoplasias, inflammatory bowel disease, and angiodysplasias. ¹⁰ Primary or metastatic GTD of the gastrointestinal tract is very infrequent. Metastatic GTN occurs in 4% of patients after evacuation of the complete mole and infrequently after other pregnancies. ^11,12 GTN usually metastasizes as choriocarcinoma because of its propensity for early vascular invasion with widespread dissemination. The most common metastatic sites are the lung (80%), vagina (30%), brain (10%), and liver (10%). ¹ Because trophoblastic tumors are perfused by fragile vessels, metastases are often hemorrhagic.

We performed a MEDLINE search using the terms “hydatidiform mole,” “ectopic molar pregnancy,” “metastatic mole,” and “gestational trophoblatic disease or neoplasm.” Our literature search did not show any cases of gastrointestinal bleeding attributable to moles.

Moreover, only a few cases were reported of molar pregnancies arising in the Fallopian tubes. ^{12 –15} No published cases of an ectopic molar pregnancy with gastrointestinal metastasis were found. In comparison, there have been several case reports of choriocarcinoma or placental site trophoblastic tumor with gastrointestinal bleeding as a presenting symptom, the cause of which being either a primary gastrointestinal malignancy, bleeding duodenal ulcer, or metastatic deposit. ^{6,16 –19} To our knowledge, this is the first report of molar pregnancy in a Fallopian tube with ileal metastasis.