Journal of Cancer Therapy, 2013, 4, 1335-1340 Published Online October 2013 ( 1335
Primary Cervical Choriocarcinoma: Case Report and the
Review of Literatures
N. Behtash, M. Karimi Zarchi*, A. A. Shamshirsaz, A. A. Shamshirsaz,
Ashrafosadt Miratashi-Yazd, N. Mehrdad
Gynecological Oncology Fellowship, Shahid Sadoughi University of Medical Science, Yazd, Iran.
Email: *
Received March 6th, 2013; revised April 6th, 2013; accepted April 14th, 2013
Copyright © 2013 N. Behtash 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.
Background: Primary choriocarcinoma of the cervix is a very rare en tity. Case: A 35-year-old patient had been admit-
ted to our hospital with vaginal bleeding for 7 months. A cervical mass measured 4 × 4 cm was palpated on the anterior
surface of the cervix. Biopsy of the lesion demonstrated extensive necrosis and was in favor of squamous cell carci-
noma. Patient was accepted as FIGO stage Ib1-2 and underwent type II hysterectomy with bilateral salpingoopherectomy
and bilateral pelvic lymph node dissection was carried out. Post operative pathologic evaluation of the surgical speci-
men evinced that the case was primary cervical choriocarcinoma. Conclusion: Primary choriocarcinoma of the cervix
should be considered in patients in their reproductive years with cervical lesion and negative cervical cytology.
Beta-hCG assay and transvaginal color Doppler sonogra ph y are usefu l for earl y diagnosi s.
Keywords: Choriocarcinoma; Cervix; Primary
1. Introduction
Choriocarcinoma was categorized into two classes: ges-
tational and non gestational. Non gestational choriocarci-
noma is a rare germ cell tumor of the ovary that only
few cases are reported in literature. Gestational choriocarci-
noma belongs to the group of the gestational tropho-
blastic disease (GTD) and usually arises in the uterine
cavity. Extrauterine chorio carcinoma is rarely seen. Saito
et al. defined following diagnostic criteria for extrau-
terine choriocarcinoma in 1965: 1) absence of disease in
the uterine cavity, 2) exclusion of coexistence of normal
intrauterine pregnancy, 3) exclusion of molar pregnancy
and 4) pathologic confirmation of disease [1]. Most of
the extrauterine choriocarcinoma reported in literature,
originates in uterine cervix [2-7]. However, there are also
reports in the literature that choriocarcinoma located in
other regions outside the uterus in genital tract such as
ovary [8], tube [9], vulva [10] and vagina [11]. Our ob-
jective was to describe the clinical and pathologic find-
ings of one case of primary cervical choriocarcinoma in a
35-year-old woman and reviews of the literatures.
2. Materials and Methods
From January 1950 to December 2008, a PUBMED/
MEDLINE search performed using the key words chori-
ocarcinoma and cervix and primary. Totally 29 publica-
tions including 78 patients existed (Table 1), which 18
are in the PUBMED/MEDLINE. In our review, we ex-
cluded all papers other than English language. We got
information regarding age, gravida and parity, symptoms
and duration, preceding pregnancy, latent period from
preceding pregnancy, location, lesion size and hCG level
(Table 2). Also type of therapy and the outcomes were
attained and mentio ned in separate table (Table 3).
3. Case Presentation
A 35-year old woman, Gravid 3, Para 2 and Abortion 1,
had been experienced irregular vaginal bleeding for 7
months when she was referred to our clinic on June 2008.
Her first and second pregnancy ended in a full-term de-
livery by caesarean section in 37 and 38 weeks of gesta-
tion in 2002 and 2004, respectively. Her third pregnancy
ended in a missed abortion at 6 week’s gestation in 2005.
She mentioned a regular menstrual cycle with 7 days
mean duration. She complained of bleeding 4 days before
her menses, and in the last month, she mentioned con-
*Corresponding a uthor.
Copyright © 2013 SciRes. JCT
Primary Cervical Choriocarcinoma: Case Report and the Review of Literatures
Table 1. Eighty-seven cases of primary cervical choriocarcinoma recorded in literature.
Study Cases Journal Language/Country
Rashbaum M* (1952) 13 (12 from l it erature) Am J Obstet Gynecol 1952; 6 4: 451-5 English/Amer i ca
Saito M (1965) 18 (18 from literature) World Obstet Gynecol 1965; 17: 459-84 Japanese/Japan
Minegishi (1965) 6 Sanfujinka Jissai 1965; 14: 763 -7 Japanese/Japan
Koga (1966) 1 J Jpn Obstet Gynecol Soc 1966; 13: 245-9 Japanese/Japan
Ooguchi (1966) 7 J Jpn Obstet Gynecol Soc 1966; 18: 1083-92 Japanese/Japan
Danek (1969) 1 Minerva Ginecol 1969; 21: 1707-11 Italian/Italy
Momose (1970) 1 Obstet Gynecol Ther 1970; 21: 468-71 Japanese/Japan
Pavlica (1971) 1 Zentralbl Gynakol 1971; 93: 72- 4 German/Germany
Tsukamoto* (19 80) 1 Gynecol Oncol 1980; 9: 99-107 English/Japan
Tripathi* (1982) 1 Br J Obstet Gynecol 1982; 89: 267-9 English/India
Meriah (1983) 1 J Gynecolo Obstet Biol Reprod(Paris) 1983; 12: 51 9-24 French/France
Martin* (1983) 1 Am J Obstet Gynecol 1983; 147: 343-4 English/Americ a
Bogdanowicz (19 84) 1 Ginekol Pol 1984; 55: 527-30 Polish/Poland
Bhalla* (1987) 1 Indian J Pathol Microbiol 1987; 3 0: 51-3 English/India
Tsai*(1988) 4 Asia Oceania J Obstet Gynaecol 1988; 14: 285-9 2 English/Taiwan
Ben-Chetrit* (1990) 1 Am J Obstet Gynecol 19 90 ; 1 63: 1161-3 English/Israel
Lee* (1992) 1 Acta Obstet Gynecol Scand 1992; 71: 4 79-81 English/ Taiwan
Heyn (1993) 1 Geburtshilfe Frauenheilkd 1993; 53: 498-500 German/Germany
Herts* (1993) 1 J Ultrasound Med 1993; 12: 59-62 English/America
Abboud (1994) 1 J Gynecol Obstet Biol Reprod (paris) 1994; 23: 149- 51 French/France
Al Hassani* (1995) 1 Trop Geogr Med 1995; 47: 308-9 English/Qatar
Yahata* (1997) 1 Gynecol Oncol 1997; 64: 274-8 English/Japan
Lema* (1997) 1 East Afr Med J 1997; 74: 600-2 English / Malawi
Baykal* (2003) 1 Gynecol Oncol 2003; 90: 667-9 English/Turkey
Huang (2004) 2 Zhong Nan Da Xue Xue B ao Yi Xue Ban 2004; 29: 108-9 Chineese/China
Roopnarinesingh* (2004) 1 Ir Med J 2004; 97: 14 7-8 English/Irland
Peko (2005) 1 Med Trop 2005; 65: 498 French/France
Fu* (2007) 4 Int J Gynecol Cancer 20 07; 17: 715-719 English/China
Kairi-Vassilatou* (2007) 1 Int J Gynecol Cancer 2007; 17: 921-925 English/Greece
tinuous spotting for one month. On physical examination,
necrotic hemorrhagic polypoid mass protruding into cer-
vical canal, approximately 4 × 4 cm, was palpated. Va-
ginal fornices were normal. Digital examination demon-
strated no parametrial extension. Colposcopy was done
and punch biopsy was taken. During biopsy significant
bleeding from the lesion occurred and because of ob-
scured vision only eight specimens were excised. Patho-
logy reported that six of eight specimens were necrotic
tissue with atypia in favor of squamous cell carcinoma
and two specimens demonstrated dysplasia.
On investigation, Biochemistry profile and chest X ray
(postero-anterior) were within normal limits. Computed
tomography of the abdomen and pelvic were normal. No
metastasis was found. Abdominal sonography showed
normal structures with uterus in normal size and normal
endometrium. Intravenous pyelography, cystoscopy and
proctoscopy were within normal as well. Then, the pa-
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Primary Cervical Choriocarcinoma: Case Report and the Review of Literatures 1337
Table 2. Clinical featured of the patients with primary cervical choriocarcinoma in the literature.
Study Age Gravida
and Para Symptoms Preceding
Latent Period
form Preceding
Pregnancy LocationLesion Size Serum or Urine
Rashbaum (1952) 38 G6P4
Vaginal Bleeding +
Abdominal pain
(2 weeks)
abortion 11 months Posterior
wall 6 * 8 * 4 cm
(macrography) -
Tsukamoto (198 0) 49 G4P2
Vaginal spotting +
lower abdominal
pain (2 weeks)
abortion 36 months - 4.5 * 3.5 cm
angiography) 8000 U/L
Tripathi (1982) 28 G2P2 Menorrhagia (7
months) Term
delivery 7 months - 6 * 5 * 2.5 cm
(macrography) 540,000 i. u/24 h
Martin (1983) 35 G1P1 Menorrhagia
(3 months) Term
delivery 12 months - 4.0 * 3.5 * 2.5 cm
(macrography) Positive but not
Bhalla (1987) 30 G4P3
Vaginal bleeding
(7 weeks) Induced
abortion 12 months - 6 * 5 * 4 cm
(macrography) Positive with
dilution 1/32
Case 1 46 G7P4 Menorrhagia with
occasionally massive
bleeding (2 months)
abortion 48 months - 9 * 7 * 4 cm
(macrography) 162,250 mIU/ml
Case 2 43 G1P1 Hemorrhagic shockTerm delivery29 years Posterior
wall 3 cm Positive but not
Case 3 29 G4P3 Menorrhagia
(several months) Induced
abortion 48 months - - Positive but not
Case 4 53 G7P5 - Hydatidiform
mole 3 months - - -
Ben-Chetrit (199 0) 33 G3P3 Menorrhagia
(1 month) Term delivery12 months - 0.5 * 1 cm (pelvic
examination) 3 2 mIU/ml
Lee (1992) 29 G2P1 Profuse vaginal
bleeding (6 hours)Hydatidiform
mole 9 months Left wall8.6 * 7.7 cm
(sonography) 150,580 mIU/ml
Herts (1993) 47 G1P1 Heavy vaginal
bleeding (6 days) Term delivery18 years - 5 cm
(sonography) 109,870 mIU/ml
al Hassani (1994) 40 G3P3(a) Post-coital bleeding
(2 months) Term delivery- Anterior
lip 2.5 * 2 cm
(macrography) 244 IU/L
Yahata (1997) 38 G4P2 Menorrhagia
(2 weeks) Term delivery4 months - 6 * 5 cm (Doppler
sonography an d
MRI) 128,000 IU/L
Lema (1997) 36 G11P10
Vaginal bleeding in
28 weeks of
gestation (2 days)Term delivery24 months Posterior
lip 4 * 2 cm
(macrography) -
Baykal (2003) 54 G5P4 Postmenopausal
bleeding Term delivery17 years - 3 cm (pelvic
45,000 IU/L
after pathology
(2004) 25 G1p0
Abdominal pain and
mild vaginal
bleeding (2 months)
mole 24 months Anterior
lip 4 * 3 * 5 cm
(MRI) 4150 U/L
Case 1 46 G6P4 Vaginal bleeding
(4 months) Induced
abortion 64 months Posterior
cervix 3 * 4 cm
(macrography) 20,000 U/L
Case 2 21 G1P0 Vaginal bleeding
(8 months) Hydatidiform
mole 20 months Posterior
cervix 5 * 7 cm
(macrography) 5344 U/L
Case 3 35 G4P2 Vaginal bleeding
(1 month) Induced
abortion 47 months Posterior
cervix 2 * 3 cm
(macrography) 4000 U/L
Fu (2007)
Case 4 30 G3P2 Vaginal bleeding
(3 months) Term delivery12 months Posterior
cervix 3.4 * 2.5 cm
(sonography) 2764 U/L
(2007) 43 G7P2 Amenorrhea
(2 months) Term delivery12 months Posterior
cervix 5 * 3 cm
(macrography) 7485 mIU/ml
(a): Patient had amenorrhea 3 years before her symptoms and D & C was performed for patient, but there was no report of the pathology. So the gravida is
subject to change (G4).
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Primary Cervical Choriocarcinoma: Case Report and the Review of Literatures
Table 3. Staging, treatment and outcomes of the patients with primary cervical choriocarcinoma in the literature.
Study Treatment Outcome
Rashbaum TAH + BSO Died (3 months after diagnosis)
Tsukamoto TAH + BSO followed by 3 courses of MTX Survived without recurrence
after 9 months
Tripathi Oral MTX for 2 days followed by TAH followed by 3 days MTX
followed by repe at course of MTX after 2 weeks Survived without recurre nce
after 10 months
Martin MTX + TAH + 3 postoperative courses of MTX -
Bhalla TAH + BSO followed by 1 course of MTX Recurrence with pulmonary metastasis
(after 3 months)
Case 1 TAH + BSO followed by 4 courses MTX + actinomycin D No Recurrence on follow up
Case 2 Radical Hysterectomy (suspicious for cervical cancer) Re c u rrence with pulmonary and brain
metastasis (1 month and 2 months)
Case 3 TAH + BSO Recurrence with pulmonary metastasis
(6 months) died (14 months)
Case 4 Under Hydatidiform mole, subtotal hysterectomy + BSO performed.
Patient returned after 2 years with cervical choriocarcinoma and
pulmonary metastasis
Recurrence with pulmonary metastasis
and cervical choriocarcinoma (2 years)
Ben-Chetrit 2 course of MTX + folinic acid (patient desired future pregnan cy) Survived without recurrence
after 12 months
Lee Vaginal Hysterectomy + 4 courses actinomycin D (MTX
discontinued because of abnormal LFT) Survived without recurrence
after 20 months
Herts Vaginal Hysterectomy + Unilateral salpingo-oophorectomy Survived without recurrence
after 6 months
al Hassani TAH + BSO + chemotherapy -
Yahata 2 courses MAC followed by TAH followed by 6 cours es ACM Survived without recurrence
after 18 months
Lema TAH Missed on follow-up after surgery
Baykal Type III hysterect omy + BSO + Pelvic-paraaortic LAD followed by
EMA-CO chemotherapy -
Roopnarinesingh MTX for 8 weeks (alternate week) Survived with locally recurrence
after 12 months
Case 1 TAH + BSO followed by 2 course of ACM chemotherapy Survived without recurrence
after 17 years
Case 2 TAH followed by 2 courses of ACM after two courses of AF
performed at h e r h ome town Survived without recurrence
after 16 years
Case 3 3 courses of ACM after TAH + USO foll owed by 1 course 5-FU Survive without recurrence after 14 years
Case 4 1 course of ACM chemoth er apy following with TAH and 2 courses
of ACM Chemo t h erapy after TAH Survived without recurrence after 6 years
-: No information, ACM: Actinomycin D, cyclophosphamide and methotrexate; AF: Actinomycin D and 5-Fluouracil; BSO: Bilateral salpingo-oophorectomy;
EMA-CO: Etoposide-methotrexate-dactinomycin-cyclophosphamide-vincristine; 5-FU: 5-fluouracil; LAD: Lymphadenectomy; LFT: Liver function test; MTX:
Methotrexate; TAH: Total a bdominal hysterectomy; USO: Unilateral salpingo-oophorectomy.
tient was accepted as FIGO stage Ib1-2 cervical cancer.
One week later the patient underwent type II hysteric-
tomy with bilateral salpingoopherectomy and pelvic
lymph node dissection. At operation, a soft necrotic
hemorrhagic mass, approximately 4 × 5 cm, was found
on the anterior surface of the cervix.
On gross examination, the hysterectomy specimen
presented with a soft, brown-colored mass of 3.5 cm in
the largest diameter which was intramural and did not
involve the lumen. The tumor formed a necrotic hemor-
rhagic mass confined to the lower part of cervix without
extending to the endometrium or myometrium.
On microscopic examination sections revealed a ne-
crotic hemorrhagic tumor that is distinct from the epithe-
lial surface of both endocervix and ectocervix. So, this
distinction proved that tumor had not had epithelial ori-
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Primary Cervical Choriocarcinoma: Case Report and the Review of Literatures 1339
gin and we could exclude Squamous cell carcinoma or
adenocarcinoma. On magnification Malignant tropho-
blastic tumor composed of atypic bizarre polygonal tro-
phoblasts with nucleolated nuclei, High nuclear to cyto-
plasm ratio (N/C ratio) and atypic mitotic figures which
are strongly positive for B-HCG and pancytokeratin.
Human placental lactogen (HPL) was weakly positive in
few cells. There was also hemorrhage (Blood Lake) in
tissue and for this reason, the pathologist performed Im-
munohistochemistry of CD31 and VIII Factor to exclude
tumor with endothelial origin like angiosarcoma. Based
on these findings, choriocarcinoma was diagnosed for the
patient. An extensive sectioning of the endometrium,
myometrium, parametrial tissues and lymph nodes con-
firmed that these areas were free of neoplastic cells or
any other site of trophoblastic cells.
Patient discharged with good condition four days later
and postoperative B-hCG was 6 mIU/ml after 2 weeks of
operation. According to the FIGO staging system of
GTD, the patient was classified as stage I, but because
there was no preoperative B-hCG level we could not de-
termine the risk score of the patient [12]. The patient is
under follow-up and has had serial B-hCG measurement,
which has been declining through 4 weeks. She is doing
well without any complain so far.
4. Discussion
Gestational intrauterine choriocarcinoma has higher inci-
dence rate in Asia, Africa and Latin America in com-
parison to United States and Europe, may be due to so-
cioeconomic status [13]. However, the overall incidence
of choriocarcinoma in recent years has been decreased
parallel to improving socioeconomic conditions [13]. Pri-
mary choriocarcinoma of the cervix is an extremely rare
disease; however, most of the case reports in literatures
demonstrate that extrauterine choriocarcinoma mostly
originates in cervix rather than other sites in genital tract
[2 -7] . O ur ca se fulfilled the criteria defin ed by Saito et a l.
[1]. So, it is an ectopic choriocarcinoma originates from
the cervix.
The median age was 37 with the range of 21 - 54 year s
old (two patients were postmenopausal). 9 patients were
less than 35 years old. The latent period from preceding
pregnancy had a range of 3 - 348 months with 62% less
than or equal to 24 months. Long latency of choriocarci-
noma after the preceding pregnancy is not common [14].
Choriocarcinoma is heterochronic if the latent period is
more than two years [15]. Choriocarcinoma occurred in
our patients 3 years after the preceding pregnancy, which
was terminated by induced abortion. There was limited
data regarding the location of tumor with mostly origi-
nated from posterior cervix.
Gestational choriocarcinoma may follow any type of
pregnancy, which most preceded by molar pregnancy
(50%), 25% developed after abortion and 25% follow an
apparently normal pregnancy. The more common gesta-
tional variety is associated with a coincident and antece-
dent pregnancy. In this review primary cervical chorio-
carcinoma preceded by 50% normal pregnancy, 32%
abortion and 18% de vel o ped after hydatidi f orm mole.
There are some theories expressed for developing of
cervical choriocarcinoma [16,17]. It is possible that it can
be a metastatic tumor from a vanishing intrauterine cho-
riocarcinoma or it could be a transformation of a cervical
pregnancy. The pathologic examination ruled out these
possibilities, as we demonstrated that no trophoblastic
cell was observed in endothelium and myometriu m of the
uterus. Another theory expresses the role of therapeutic
abortion in the implantation of trophoblasts in the cervix
and lower genital tract. Trauma to the cervix during D &
C may be a predisposing factor [6]. Our case was pre-
ceded by an abortion and it supports the later theory.
There was no pathological examination of the curetted
material as this was performed in another city and we did
not have access to the pathology.
The clinical diagnosis of primary choriocarcinoma is
very difficult. In this review all patients except one,
complained of vaginal bleeding with different period of
time ranged from 6 hours to 8 months, with different
severity that one patient referred with hypovolemic shock.
Only one patient complained of amenorrhea. The predo-
minant symptom is abnormal vaginal bleeding which is
due to angiogenesis and neovasculirization of this tumor.
There are few differential diagnoses which include uter-
ine cervical pregnancy, threatened abortion, a cervical
polyp and malignancy [4]. In the present case, choriocar-
cinoma was presented with vaginal bleeding and misdi-
agnosed as malignancy. For this reason, we should con-
sider choriocarcinoma as differential diagnosis in any
patient in their reproductive years with a cervical lesion
with high vascul ari t y and pr o fuse bleeding.
How can we suspect and diagnose cervical choriocar-
cinoma? As we mentioned above, in cervical lesions in
women of child-bearing age with profuse bleeding,
measurement of serum and urinary B-hCG is useful for
diagnosis. Color Doppler transvaginal ultrasonography
and MRI with con trast detect the abundan t blood flow in
the tumor [4]. Transvaginal Doppler sonography allows
detection of tumoral vessels, which manifests high dia-
stolic blood flow and low impedance resulting in ne-
ovasculirization [4]. Contrast-enhanced MRI can demon-
strate tumoral vascularity and in cases of necrosis hy-
pointensity of the necrotic areas are perceptible [4].
Diagnosis of such a kind of cervical tumor is very dif-
ficult for pathologist both in biopsy and surgical resec-
tion. In our case, the first pathologist misdiagnosed
choriocarcinoma with squamous cell carcinoma as Ben-
Copyright © 2013 SciRes. JCT
Primary Cervical Choriocarcinoma: Case Report and the Review of Literatures
Copyright © 2013 SciRes. JCT
Chetrit [18]. Necrosis is a predominant feature of these
neoplasms, often to such an extent that the viable tissue
is obscured. So, we should consider immunohistochemi-
cal staining in extend necrotic tissue obtained from cer-
vix for more information, because there is the chance for
more limited surgery and more successful chemotherapy
for choriocarcinoma if it is diagnosed in the first step. In
our case, massive necrosis of the tumor and arising of
tumor from non-epithelial origin motivate the pathologist
to perform immunohistochemical staining. As we show-
ed the specimen was markedly positive fo r B-hCG.
The treatment of choice as in our patient was total ab-
dominal hysterectomy and bilateral salpingoopherectomy
with or without chemotherapy. In terestingly, Ben-Chetrit
and his co-worke rs in one report and Roopnarinesingh et
al. in other report, documented conservative management
of primary cervical choriocarcinoma [4,18].
5. Conclusion
In conclusion, cervical choriocarcinoma should be con-
sidered in the differential diagnosis of cervical lesions in
their child-bearing years, especially lesions with promi-
nent vascularity. A beta-hCG assay, transvaginal color
Doppler sonography or contrast-enhanced MRI may
form part of pre-operative investigation in such a patient.
A detailed correlation between histologic findings and
patient’s clinical history, lab studies and radiologic data
is always required to preclude diagnostic pitfalls and de-
signates the appropriate treatment.
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