15 June 2021: Articles
A Case of Rapid Transformation from Hydatidiform Mole to Invasive Mole: The Importance of β-hCG (Human Chorionic Gonadotropin) Serum Levels in Follow-Up Evaluation
Unusual setting of medical care, Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)Ali Budi Harsono A , Yudi Mulyana Hidayat C , Gatot Nyarumenteng A. Winarno E* , Aisyah Shofiatun Nisa E , Firas Farisi Alkaff E
Am J Case Rep 2021; 22:e931156
BACKGROUND: Gestational trophoblastic disease (GTD) is a spectrum of disorders consisting of premalignant (ie, complete [CHM] and partial hydatidiform moles [PHM]) and malignant conditions (ie, invasive moles, choriocarcinoma, placental site trophoblastic tumors, and epithelioid trophoblastic tumor). If GTD persists after initial treatment and has persistent elevated beta human chorionic gonadotropin (β-hCG), it is referred to as post-molar gestational trophoblastic neoplasia (pGTN). To date, there is no detailed information regarding how fast invasive moles can develop from CHM. However, the risk of developing any pGTN from CHM is rare within 1 month and is greatest in the first 12 months after evacuation, with most cases presenting within 6 months.
CASE REPORT: We present a case of a 46-year-old primigravida woman with rapid transformation of an invasive mole. In the beginning, the patient had a chief concern of a uterus size greater than the gestational dates. Laboratory evaluation showed high β-hCG serum level (>300 000 mIU/mL), and ultrasonography evaluation revealed a hydatidiform mole. Suction evacuation and curettage procedures were then performed. Pathology evaluation afterwards revealed a complete hydatidiform mole without any sign of malignancy. Twenty-two days afterwards, the patient came to the emergency room with vaginal bleeding. β-hCG serum level was high (53 969 mIU/mL), and ultrasonography examination showed the presence of fluid filling the uterine cavity. The patient was then diagnosed with GTN, and hysterectomy was chosen as the treatment of choice. After the surgery, her β-hCG serum level gradually reverted back to normal.
CONCLUSIONS: Invasive moles can develop less than 1 month after suction evacuation and curettage procedure for CHM. Serial β-hCG serum level evaluation according to the guideline should be performed to prevent late diagnosis, which could lead to the development of metastasis and worsen the prognosis.
Keywords: gestational trophoblastic disease, Hydatidiform Mole, Hydatidiform Mole, Invasive, Chorionic Gonadotropin, Follow-Up Studies, Middle Aged, Placenta, Pregnancy, Uterine Neoplasms
Gestational trophoblastic disease (GTD) is a spectrum of disorders consisting of premalignant (ie, complete [CHM] and partial hydatidiform mole [PHM]) and malignant conditions (ie, invasive mole, choriocarcinoma, placental site trophoblastic tumor, and epithelioid trophoblastic tumor). If GTD persists after initial treatment and has persistent elevated beta human chorionic gonadotropin (β-hCG), it is referred to as post-molar gestational trophoblastic neoplasia (pGTN) . Studies showed that the incidence of PHM is higher than that of CHM, but CHM has a more than 7 times higher risk of developing pGTN [2,3]. About 10–17% will develop into an invasive mole, which is a benign tumor that arises from myometrial invasion via direct extension through tissue or venous channels. Although it is a benign tumor, the mortality rate is 15% .
To date, there is no detailed information regarding how fast an invasive mole can develop from CHM. However, the risk of developing any invasive mole from CHM is low within 1 month and is greatest in the first 12 months after evacuation, with most cases presenting within 6 months [3,5]. In this case report, we present a rare case of an invasive mole that developed 22 days after the evacuation of CHM. This case report highlights the importance of serial β-hCG follow-up evaluation, as recommended in the guidelines .
A 46-year-old primigravida woman was referred to our outpatient clinic in October 2020 with a chief concern of a uterus size greater than the gestational dates. Her last date of menstruation was 8 weeks ago, and the pregnancy test showed a positive result. The patient never had a miscarriage or underwent an abortion. Vital signs were unremarkable. Physical evaluation revealed an enlarged uterus proportional to 16 weeks of pregnancy. Laboratory evaluation showed elevated β-hCG serum level (>300 000 mIU/mL), while other parameters were within normal ranges. Ultrasonography (USG) evaluation revealed a hydatidiform mole (Figure 1); therefore, suction evacuation and curettage were performed. After the procedures, USG reevaluation showed that the hydatidiform mole was completely removed (Figure 2). The samples were sent to the pathology anatomy lab for macroscopic and microscopic evaluation, which later revealed a complete hydatidiform mole without any sign of malignancy (Figures 3, 4).
The patient was scheduled for follow-up β-hCG serum level evaluation on the 7th, 14th, and 21st days after the procedure. However, the patient did not comply with the follow-up schedule. The patient came on the 9th day after the procedure, where follow-up evaluation showed that β-hCG serum levels fell to 9838 mIU/mL. Because of the significant reduction of the β-hCG serum levels, the patient decided to skip the evaluation on the 14th day. On the 22nd day, the patient came to the emergency room with severe vaginal bleeding. The USG examination showed the presence of fluid in the uterine cavity (Figure 5). The hemoglobin level had fallen to 6 mg/dL, and the β-hCG serum level drastically increased to 53 969 mIU/ mL. The patient was then diagnosed with GTN. The chest X-ray evaluation showed no sign of distant metastases.
Based on several considerations and discussion with the patient, hysterectomy was chosen as the treatment of choice. Hysterectomy was performed 2 days later, after the hemoglobin level was increased to 8 mg/dL. The specimen was then sent to the pathology anatomy lab for evaluation, which later established the diagnosis of an invasive mole (Figures 6, 7). After the surgery, the patient was scheduled for serial β-hCG serum level evaluation. The β-hCG serum level were 355 mIU/mL on the 7th day, 115 mIU/mL on the 14th day, and 62 mIU/mL on the 21st day.
The patient did not comply with the follow-up schedule for serial β-hCG serum level evaluation. Therefore, we cannot provide data on exactly when the β-hCG level started to rise again.
Invasive moles can develop in less than 1 month after suction evacuation and curettage procedures for CHM. Serial βhCG serum level evaluation according to the latest guidelines should be performed to prevent late diagnosis, which could lead to the development of metastasis and worsen the prognosis. Further research is needed to understand the pathological mechanism of the transformation and factors that contribute to the rapidity of the transformation.
FiguresFigure 1.. The USG Examination before curettage. Figure 2.. The USG Examination after curettage. Figure 3.. Macroscopic picture of complete hydatidiform mole suction evacuation and curettage procedures showed mole bubbles. Figure 4.. Microscopic image of complete hydatidiform mole showing chorialis villi experiencing hydrophilic degeneration of avascular scalloped form and accompanied by the formation of cisterns lined with proliferative cytotrophoblast cells. Figure 5.. The USG Examination after recurrence. Figure 6.. Macroscopic image of uterus with invasive mole in the fundus. Figure 7.. Microscopic image of invasive mole showing chorialis villi that are lined with hyperplastic trophoblast cells and have invaded the myometrium. The villous fibrovascular stroma is partly hydrophilic, with avascular degeneration.
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