Primary choriocarcinoma of the colon: a case report and review of the literature.

1.9 MB·English

by  Jiang, Lun

Jiangetal.WorldJournalofSurgicalOncology2013,11:23
http://www.wjso.com/content/11/1/23 WORLD JOURNAL OF
SURGICAL ONCOLOGY
CASE REPORT Open Access
Primary choriocarcinoma of the colon: a case
report and review of the literature
Lun Jiang, Jing-Tao Wu* and Xin Peng
Abstract
Choriocarcinoma usually arises in theuterusand gonads. Primary choriocarcinoma (PCC)in an extragenital organ is
rare. When it occurs in thegastrointestinal tract, thestomach is the most common site. Only 12 cases ofPCC of the
colon have been reported inthe world literature.Most cases were associated withadenocarcinoma. We report the
case of a 36-year-old man withPCC of thecolon and review theclinical characteristics ofpreviously documented
cases.
Keywords: Adenocarcinoma, Chemotherapy, Choriocarcinoma, Colon, Syncytiotrophoblastic differentiation
Background in the normal range. His serum beta human chorionic
Choriocarcinoma is a highly malignant tumor of tropho- growthhormone (β-HCG)levelwas3.38mIU/ml.Acol-
blastic cells that most often arises in the female genital onoscopyrevealedayellowtumorinthecolon(Figure2)
tract. It is considered to be pertinent to molar preg- and endoscopic biopsy findings suggested a poorly dif-
nancy, normal or ectopic pregnancy and abortion. When ferentiated adenocarcinoma. After two days, a colectomy
itoccurs inmale patients,the testis isthe most common was performed. The tumor located in the ascending
site. Choriocarcinoma of extragenital origin is found in colon measured 4 cm × 5 cm and penetrated the serosa
the retroperitoneum or the mediastinum, or intracalvar- and the mesocolic fat, with 12 adjacent enlarged lymph
ium. Primary choriocarcinoma (PCC) originating in the nodes. The pathologic findings showed that the tumor
colon is extremely rare and the prognosis is usually invaded the serosa of the intestinal wall and was com-
poor. posed of syncytiotrophoblastic cells, cytotrophoblast-like
cells and intermediate trophoblastic cells. Necrosis and
hemorrhage were also noted in the mass. Immunohisto-
Case presentation
chemical staining was positive for HCG and negative for
A 36-year-old male patient visited the local hospital for cytokeratin 7, cytokeratin 20, villin, caudal type homeo-
the chief complaint ofdiscomfortoftheupperabdomen. box 2, α-fetoprotein and CD30 (Figure 3). Metastasis
An appendectomy was performed under the clinical was found in the liver and in eight of the excised lymph
diagnosis of acute appendicitis. During the operation, a nodes.
tumor was observed in the colon. The operation was Systemic chemotherapy using bleomycin, etoposide
aborted because of an inability to remove the tumor. and platinum was performed. Chemotherapy engaging
Two days later, a computed tomographic (CT) scan of etoposide phosphate 100 mg, cisplatin 30 mg, bleomycin
the abdomen revealed the mass arising from the ascend- 18 mg was initiated. The β-hCG level increased from
ing colon. The regional enlarged lymph nodes and me- 3.38 to 10,000 mIU/ml after the first course of chemo-
tastasesintheliverwere noted (Figure 1). therapy, and CTshowed another mass in the liver. After
The patient was transferred to our hospital for treat- three cycles of chemotherapy, his β-hCG level remained
ment. Tumor markers, including carcinoembryonic anti- high. Because of the progression of disease, the patient
gen, cancer antigen 19–9 (CA 19–9) and CA 125, were and his family abandoned other alternative regimens of
chemotherapy.
*Correspondence:[email protected]
DepartmentofMedicalImaging,SuBeiPeople’sHospitalofJiangSu
Province,MedicalSchoolofYangzhouUniversity,No.98NanTongWest
Road,Yangzhou,JiangsuProvince225001,China
©2013Jiangetal.;licenseeBioMedCentralLtd.ThisisanOpenAccessarticledistributedunderthetermsoftheCreative
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reported, 61.5% of patients presented with the tumor in
theproctosigmoid;thetumorwaslocatedintheascend-
ingcoloninthreepatients(Table1).
The pathogenesis of primary extragenital choriocar-
cinoma is controversial. Several hypotheses have been
proposed, including that it develops from retained
primordial germ cells that migrated abnormally during
embryonic development [16]; metastasis from a latent
primarylesioninthegenitalia[4];andtheretrodifferen-
tiation of pre-existing colonic carcinoma [8]. Among
them, the retrodifferentiation theory from pre-existing
colonic adenocarcinoma is well accepted. Given that
PCC of the colon associated with adenocarcinoma
accounts for 69.2% of reported cases, this theory
is considered to be logical. However, concurrent
Figure1Computedtomography.(A)Notethethickenedwallof
theascendingcolon(shortarrow)andenlargedlymphnodesinthe
mesentery(longarrow).(B)Thehepaticlesionmanifested
ring-shapedenhancement(arrow).
Discussion
Choriocarcinoma most commonly arises in tropho-
blastic tissue following gestational events such as molar
pregnancy, normal or ectopic pregnancy, and abortion.
When the tumor occurs in men, the gonads are the
most common sites. Choriocarcinoma of extragenital
origin has been reported in the retroperitoneum [1],
mediastinum [2], and intracalvarium (especially in the
pineal gland [3]). When it occurs in the gastrointestinal
tract, the stomach is the most common site [4]. PCC
originating in the colon is extremely rare, with only 12
documented cases. In the literature, the mean age of Figure2Endoscopicbiopsy(A)Colonoscopyrevealedacircular
presentation with PCC of the colon is 51.4 years, and yellowtumorintheascendingcolon.(B)Anendoscopicbiopsy
revealedatypicalepithelium(hematoxylinandeosin×100).
the ratio of female to male is 1.6:1. Of the cases
Jiangetal.WorldJournalofSurgicalOncology2013,11:23 Page3of5
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resection followed by adjuvant chemotherapy was the
besttreatmentofPCCofthelung[16].Forpatientswith
widespread metastasis, resection has proven ineffective
in prolonging survival. In patients without metastasis, a
combination of radical surgery and appropriate adjuvant
chemotherapy contributed to their long-term survival
[4]. The chemotherapy modality for primary extragenital
choriocarcinoma has not been established; most treat-
ment options are based on experience. The chemother-
apy regimens for choriocarcinoma and adenocarcinoma
are different. Chemotherapy for both the choriocarcin-
oma and the colonic adenocarcinoma is most logical
according to a study by Kubosawa et al. [4] – their pa-
tient showed thelongest survival.
The correlation between the β-hCG level and the
effectiveness of treatment has notbeen detailed in most
previous reported cases. β-hCG did not decrease to
normal levels after surgery, implying that micrometa-
static disease existed [17]. The reduction of β-hCG in
the serum indicates response to treatment.After the re-
section of the primary lesion, the serum β-hCG level in
our case did not decrease to normal range. Abdominal
CT showed hepatic lesions when the serum β-hCG
increased. We suggest the serum β-hCG level could be
used as a marker when assessing the effectiveness of
treatment.
Conclusions
The prognosis of PCC of the colon is poor. In our ana-
lysis of the literature, the median survival time was less
Figure3Pathologicfindings.(A)Pathologicfindingsofthe
than 1 year. PCC of the colon is usually not identified
resectedspecimenrevealedpolynucleargiantcells.(B)
Immunohistochemicalstainingwaspositiveforhumanchorionic until the tumor has generalized metastasis. The great
growthhormone. majority ofpatientsdie from liverfailure.
Consent
adenocarcinoma was not present in our patient nor in Written informed consent wasobtained from thepatient
two of the previous cases [10,13]. An explanation for forpublicationofthismanuscript.
choriocarcinoma unaccompanied by adenocarcinoma
could be that the tumor arises directly from malignant
Abbreviations
changeintheectopicchorionortotipotentcells[13]. β-HCG:betahumanchorionicgrowthhormone;CA:cancerantigen;
Thepreoperative diagnosisofPCCofthe colon is poor. CT:computedtomography;PCC:primarychoriocarcinoma.
In our case, the patient was assessed as having metastatic
colonic cancer. An endoscopic biopsy supported this pos- Competinginterests
Theauthorsdeclarethattheyhavenocompetinginterests.
sibility because of an insufficient specimen. However, on
postoperative biopsy, he was re-diagnosed with PCC of
thecolon.Thediagnosisofprimaryextragenitalchoriocar- Authors’contributions
Jing-TaoWuandLunJiangproposedthestudy.Jing-TaoWu,LunJiangand
cinoma should firstly rule out any abnormal findings in
XinPeng-wrotethemanuscript.LunJiangandXinPengcontributed
the uterus or genitalia. The presence of cytotrophoblast equallytothiswork.Allauthorsreadandapprovedthefinalmanuscript.
andsyncytiotrophoblastonbiopsy,thepresenceofβ-hCG
immunoreactivecells,andelevationofserumβ-hCGlevel Acknowledgements
confirmthediagnosis. WethankJunSunforprovidingtheoriginalCTimages.
The majority of patients with gastric choriocarcinoma
Received:8August2012Accepted:24December2012
are treated surgically. Umemori et al. found that Published:28January2013
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Table1Clinicalcharacteristicsofprimarychoriocarcinomaofcolon n
c
o
Case Sexandage(years) Location HCG Metastasesonadmission Associatedwith Treatment Survivaltime log
(mIU/ml) adenocarcinoma? y
2
0
ParkandReid1980[5] F/49 S NS Liverlung Y Surgery 1month 13
,
1
Nguyen1982[6] M/74 S 400 None Y Surgery 10weeks 1
:2
OrdonezandLuna1984[7] F/35 Cecum 1,612 Regionallymphnodes,liver Y Surgery 2months 3
Kubosawaetal.1984[8] F/50 S 230,000 None Y Surgery 4months
Metzetal.1985[9] F/42 S 154,000 Regionallymphnodes,liver,lungspleen Y None
LindandHaghighi1986[10] M/42 A 610,000 Liver N Surgery 1month
Tokisueetal.1996[11] F/29 R 49,000 Lung Y Surgery,chemotherapy 11months
Kiranetal.2001[12] M/68 R 700,000 Regionallymphnodes,liver Y None
Leetal.2003[13] M/73 A 146,000 Liver,lung,brain N None 10days
Verbeeketal.2004[14] F/54 R 6,831(P) Liver,lung Y Surgery,chemotherapy 8months
Froylichetal.2010[15] F/57 Descendingcolon 13,000(P) Lung N Surgery,chemotherapy 16months
Haradaetal.2012[4] F/58 S 2,420 None Y Surgery,chemotherapy Morethan60months
Presentcase2012 M/38 A 3.38 Regionallymphnodes,liver N Surgery,chemotherapy Morethan6months
A:ascendingcolon;C:cecum;F:female;M:male;NS:datanotshown;P:postoperatively;R:rectum;S:sigmoidcolon.
P
a
g
e
4
o
f
5
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Citethisarticleas:Jiangetal.:Primarychoriocarcinomaofthecolon:a
casereportandreviewoftheliterature.WorldJournalofSurgical
Oncology201311:23.
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