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CASE REPORT
Year : 2022  |  Volume : 18  |  Issue : 3  |  Page : 827-830

Cervical gestational trophoblastic neoplasia: A rare form of gestational trophoblastic disease


Department of Obstetrics and Gynaecology, King George Medical University, Lucknow, Uttar Pradesh, India

Date of Submission17-Jul-2020
Date of Decision20-Dec-2020
Date of Acceptance04-May-2021
Date of Web Publication25-Jul-2022

Correspondence Address:
Shyam Pyari Jaiswar
Department of Obstetrics and Gynaecology, King George Medical University, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_959_20

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 > Abstract 


Gestational trophoblastic neoplasia (GTN) is a rare disease and is characterized by an abnormal proliferation of trophoblastic cells of placenta. Since majority of them are chemotherapy sensitive, they are one of the highly curable cancers. However, due to its varied presentation, clinicians still face many challenges in its diagnosis and management. We present a case of 26-year-old woman, P0 + 3 (previous 3 abortions) who presented to us with a history of vaginal bleeding postuterine evacuation after 2 months of incomplete abortion. After clinical, radiological assessment and high human chorionic gonadotropin (hCG) titers, the patient was diagnosed as a case of cervical GTN. Risk assessment by the WHO prognostic scoring system was done and “Low Risk” was assigned to her. She was given total nine cycles of single agent chemotherapy including two consolidation cycles after normalization of hCG titers which patient tolerated well and remained asymptomatic.

Keywords: Cervical gestational trophoblastic neoplasia, gestational trophoblastic neoplasia, per vaginum


How to cite this article:
Jaiswar SP, Agrawal M, Deo S, Saad S. Cervical gestational trophoblastic neoplasia: A rare form of gestational trophoblastic disease. J Can Res Ther 2022;18:827-30

How to cite this URL:
Jaiswar SP, Agrawal M, Deo S, Saad S. Cervical gestational trophoblastic neoplasia: A rare form of gestational trophoblastic disease. J Can Res Ther [serial online] 2022 [cited 2022 Aug 10];18:827-30. Available from: https://www.cancerjournal.net/text.asp?2022/18/3/827/348235




 > Introduction Top


Gestational trophoblastic neoplasia (GTN) is a rare disease and is characterized by an abnormal proliferation of trophoblastic cells of placenta. Although it is a rare tumor with high cure rates, but owing to its highly metastatic behavior, it can turn out to be lethal neoplasia with widespread dissemination. Hence, it is of utmost importance to establish the diagnosis early with initiation of effective treatment on time to achieve favorable outcome. Incidence of GTN is 1:40,000 pregnancies.[1] Cervical GTN is even rarer form of this spectrum of neoplastic gestational trophoblastic disease. Early diagnosis and timely initiation of antineoplastic treatment of this metastatic disease could be lifesaving and also aids in improving the obstetric history of the patient. Our patient presented with bleeding per vaginum with already having had attempted uterine evacuation twice. Early diagnosis and prompt initiation of chemotherapy has proved miraculous in the regression of the disease and survival of the patient.


 > Case Report Top


A 26-year-old female, G3P0 + 2 (previous 2 spontaneous abortions), presented to our outpatient department with chief complaint of bleeding per vaginum since 20-day postevacuation which was done at some local hospital. The patient had previous two abortion at approximately 2.5 months of gestation 2 years and 1 year back, respectively. She resumed her normal menses after the abortion without any menstrual complaint. Elaborating the current pregnancy, according to the patient, she had an amenorrhea of 2 months with urinary pregnancy test positive which was done at home. Patient experienced bleeding per vaginum, for which she visited some local hospital. The patient was diagnosed with incomplete abortion, for which uterine evacuation was done. The patient remained asymptomatic for 7 days postevacuation after which she again started experiencing bleeding per vaginum. She was referred to district hospital where ultrasonography (USG) was done, which revealed endometrial canal full of remnants of gestational product suggestive of incomplete abortion and again uterine evacuation was done. Unfortunately, in both the attempts of uterine evacuation, no tissues were sent for histopathological examination. The symptoms continued to persist even after second uterine evacuation after which the patient reported to us for further management in a tertiary center.

On examination, the patient was well oriented, afebrile with mild anemia. Her vitals were within normal limits, weight being 52 kg and height was 160 cm. No cardiovascular and respiratory system abnormalities were detected. Abdomen was soft, nontender, and not distended. Per speculum examination revealed a fleshy polypoidal growth of 4 cm × 5 cm replacing whole of the cervix [Figure 1]. Vagina seems apparently healthy. Per vaginum examination: There was a 4 cm × 5 cm fleshy, polypoidal mass which was replacing whole of the cervix. Only a thin rim of cervix was appreciated at 12 o' clock position. The mass was bleeding on touch. Uterus was retroverted, bulky and mobile, bilateral fornices were nontender and free.
Figure 1: On per speculum examination – a 4 cm × 5 cm polypoidal fleshy mass visualized

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Investigations done for this patient were:

Patient's urine pregnancy test was done at the time of admission which was positive.

USG– [Figure 2] showed an ill defined heterogeneous heteroechoic lesion noted in the cervical canal of size 5.3 cm ×5.1 cm×3.3 cm with some cystic changes showing minimal increased vascularity on color Doppler most likely suggestive of a cervical GTN. Uterine cavity was empty with endometrial thickness of 8 mm.
Figure 2: A heterogeneous lesion noted in the cervical canal with some cystic changes

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Her beta human chorionic gonadotropin (hCG) level came out to be 35450.0 mIU/ml. Hemoglobin – 9.5 g/dl, thyroid function test – within normal limit, chest X-ray – within normal limit, and negative for any metastasis.

Treatment

Based on the clinical and radiological findings and laboratory test for hCG, a diagnosis of cervical GTN was established. The patient was staged as per the WHO Risk Prognostic Scoring System as adapted by International Federation of Obstetrics and Gynaecology (FIGO). She was assigned “Low Risk” with a score of “5.” After all the baseline examination for hemogram, serum creatinine, serum sodium, serum potassium, and liver function test, the patient was started on single agent regimen for low-risk GTN with injection methotrexate 1 mg/kg (52 mg) on days 1, 3, 5, and 7 with folinic acid rescue of 15 mg per oral on days 2, 4, 6, and 8 at every 14 days interval.

Outcome and follow-up

The patient had drastic improvement (both clinical and biochemical parameters improved) after just one cycle of single agent chemotherapy with levels of beta hCG dropping down to 2867.0 mIU/ml from 35450.0 mIU/ml and near complete disappearance of polypoidal cervical mass [Figure 3], [Figure 4], [Figure 5]. However, the patient had her liver function test deranged with serum bilirubin reaching up to 3.1 mg/dl owing to hepatotoxicity of methotrexate. The patient was warned to avoid alcohol or any herbal supplements that may increase the risk of hepatotoxicity. Treatment was continued with the first line of chemotherapy after 25% dose reduction of methotrexate.[2] The patient was well compliant with the reduced dose of methotrexate, and she tolerated the dose well with the liver enzymes ranging within normal limits throughout the therapy. The cycles continued until the beta-hCG reached 3.17 mIU/ml (<5 mIU/ml) after which two more cycles of consolidation were given, the last being in January 2020. Since then, the patient is on monthly follow-up with beta-hCG and is asymptomatic and doing well.
Figure 3: (a) During first cycle of chemotherapy. (b) After first cycle of chemotherapy

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Figure 4: Showing after second cycle of chemotherapy

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Figure 5: Showing normal looking healthy cervix during subsequent chemotherapy cycles

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 > Discussion Top


Diagnosis of cervical mass as cervical GTN is confusing as it can be easily misdiagnosed as advanced cervical carcinoma because of its morphological similarity, but high titers of beta hCG generally shift the balance more toward a GTN, leading to accurate diagnosis and correct treatment.

Although malignant gestational trophoblastic diseases are highly curable malignancies, their treatment depends on the subtypes of GTN. The subtypes are – invasive mole, choriocarcinoma, placental site trophoblastic tumor (PSTT), and epithelioid trophoblastic tumor (ETT). The first two subtypes are chemotherapy sensitive with significantly high beta hCG titers, but the last two are relatively chemotherapy resistant, so primary treatment is surgery.[3] Furthermore, in the past two subtypes, beta-hCG is generally not raised beyond 2500 mIU/ml.

The decision of chemotherapy over surgery was undertaken based on the high beta-hCG titers which inclined the diagnosis more toward a chemosensitive tumor rather than PSTT or ETT which benefitted the patient and helped her to remain disease free with no recurrences.

Jordan et al.[4] published a report differentiating squamous cell carcinoma cervix and ETT, wherein the patient presented with a cervical mass and raised hCG on which diagnosis of both squamous cell carcinoma cervix and nonmetastatic GTN was made. She was treated unsuccessfully with concurrent intravenous cisplatin and pelvic radiation and also single agent intravenous methotrexate. Review of cervical biopsy with immunohistochemistry and genotyping changed the original diagnosis to ETT. The patient died despite multiple salvage chemotherapies owing to the delayed diagnosis.

Nikolic et al.[5] highlighted the importance of early diagnosis of a poorly differentiated adenocarcinoma cervix with dominant choriocarcinoma pattern. They suggested that early and proper diagnosis of nonmetastatic choriocarcinoma of nongestational origin in coexistence with cervical cancer is curable and can have good prognosis.

Although cervical GTN is a very rare form of neoplastic GTD, recurrent history of evacuation with a fleshy growth at the involved site together with increased beta hCG titers gives a clue to the clinician to establish the diagnosis and to clarify the conundrum.

As for histological confirmation, tissue is infrequently available for the study, measurement of serum beta hCG, and clinical findings (rather than histological specimen) is used to diagnose and treat this malignancy.

Looking at the size of tumor and its rapid growth, at times, clinicians get predilected to treat it with multidrug therapy. FIGO cancer report 2018 also mentions that “Higher risk of 5–6” are at increased risk of resistance and the use of multiagent chemotherapy can be considered. However, as the results with single agent have shown to be very effective, it may guide the clinicians to effectively use the single agent therapy for the treatment of the same without adding undue toxicity related to the use of multidrug regimen to the patient.

After uterine evacuation, tissues should be necessarily sent for histopathological examination in order to seize the disease in its early stage and prevent any unwanted delay, leading to the dissemination of the disease.

Despite advances and automation in the tools used for the diagnosis of the disease, the traditional method of per speculum examination should never be omitted as at times it is a direct pointer of a particular disease and helps in setting up the diagnosis.

Hence, the key to management of such a highly curable malignancy lies in establishing the exact diagnosis and timely initiation of the precise treatment depending on the diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
Verma Y. Gestational trophoblastic disease: Dilemma and updates. Onco Exp 2017;3:01-09.  Back to cited text no. 1
    
2.
Floyd J, Mirza I, Sachs B, Perry MC. Hepatotoxicity of chemotherapy. Semin Oncol 2006;33:50-67.  Back to cited text no. 2
    
3.
Horowitz NS, Goldstein DP, Berkowitz RS. Placental site trophoblastic tumors and epithelioid trophoblastic tumors: Biology, natural history, and treatment modalities. Gynecol Oncol 2017;144:208-14.  Back to cited text no. 3
    
4.
Jordan S, Randall LM, Karamurzin Y, Ward P, Lin F, Brewster W, et al. Differentiating squamous cell carcinoma of the cervix and epithelioid trophoblastic tumor. Int J Gynecol Cancer 2011;21:918-22.  Back to cited text no. 4
    
5.
Nikolic B, Curkovic A, Dikic SD, Mitrovic A, Kuzmanovic I, Arandjelovic A, et al. Cervical poorly differentiated adenocarcinoma with dominant choriocarcinoma pattern – A case report. Vojnosanit Pregl 2015;72:651 3.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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