Pure choriocarcinoma of testis with rare gingival and skin metastases.
ABSTRACT A 22-year-old man presented with complaints of gingival and skin lesions. Physical examination revealed the presence of two nodular lesions, one over the sternum, 3 cm in size, and another, on the right side of chin, 1 cm in size. There was another fleshy soft tissue deposit over the left lower gingiva, in the oral cavity. He had noticed these lesions ten days prior to his visit to the hospital. In addition, there was left testicular non-tender swelling which had been present for two months, but was not investigated. Fine-needle aspiration cytology from skin and gingival lesions was suggestive of metastatic deposits. Patient underwent left high orchidectomy, and histopathological examination was consistent with the diagnosis of pure choriocarcinoma. Although rare, cases of testicular neoplasms and especially choriocarcinoma of the testis leading to skin metastases have been reported, but case reports of choriocarcinoma of testis metastatic to gingiva have been reported exceptionally in the English literature. We report this unique case of a young man with pure choricarcinoma of testis with unusual gingival and skin metastases.
- SourceAvailable from: ncbi.nlm.nih.gov[show abstract] [hide abstract]
ABSTRACT: BackgroundTesticular cancer, like other histopathologic types, commonly metastasizes to the lungs, liver, and brain. Spread to soft tissue, however, is rare with only four cases with seminoma reported. However, one case with metastasis of testicular immature teratoma to soft tissue was documented previously. Case DescriptionWe report the case of a 38-year-old man with recurrent immature teratoma of the testis who presented with a painless soft tissue mass in the left thigh previously treated with standard chemotherapy. After removal of the soft tissue mass, his serum alpha-fetoprotein level had returned to the normal range. Literature ReviewTo our knowledge, this is the second case of immature teratoma of the testis metastasized to soft tissue. Purposes and Clinical RelevanceWe suggest that for a man with testicular cancer who has a soft tissue mass, metastasis of soft tissue from testicular cancer and other solid malignancies should be considered in the differential diagnosis of a soft tissue mass together with primary soft tissue sarcoma.Clinical Orthopaedics and Related Research 04/2012; 468(9):2541-2544. · 2.79 Impact Factor
Singapore Med J 2007; 48(3) : e77
C a s e R e p o r t
A 22-year-old man presented with
complaints of gingival and skin lesions.
Physical examination revealed the presence
of two nodular lesions, one over the sternum,
3 cm in size, and another, on the right side
of chin, 1 cm in size. There was another
fleshy soft tissue deposit over the left lower
gingiva, in the oral cavity. He had noticed
these lesions ten days prior to his visit to
the hospital. In addition, there was left
testicular non-tender swelling which had
been present for two months, but was
not investigated. Fine-needle aspiration
cytology from skin and gingival lesions was
suggestive of metastatic deposits. Patient
underwent left high orchidectomy, and
histopathological examination was consistent
with the diagnosis of pure choriocarcinoma.
Although rare, cases of testicular neoplasms
and especially choriocarcinoma of the
testis leading to skin metastases have been
reported, but case reports of choriocarcinoma
of testis metastatic to gingiva have been
reported exceptionally in the English
literature. We report this unique case of
a young man with pure choricarcinoma
of testis with unusual gingival and skin
Keywords: choriocarcinoma, gingival
metastases, gingiva, skin metastases, testicular
choriocarcinoma, testicular tumour
Singapore Med J 2007; 48(3):e77–e80
Although every malignancy can lead to skin metastasis,
its incidence of occurrence is as low as 0.7%–9%.(1,2)
Rarely, skin metastasis represents the first evidence
of malignancy and are usually seen as a late event in
advanced cancer.(1,2) Pure testicular choriocarcinoma is
a rare germ cell neoplasm, accounting for less than 3%
of all the testicular neoplasms.(3) It usually metastasises
to the lungs, liver and brain.(3) The most common
primary sites of metastasis to the oral region are the
breast, lung, kidney, bone and colon.(4) Similar to skin
Bhatia K, Vaid A K, Rawal S, Patole K D
Cancer Institute and
Rohini Sector 5,
Bhatia K, MBBS
Vaid AK, MBBS,
Rawal S, MBBS,
Dr Ashok K Vaid
Tel: (91) 11 2705 1011
Fax: (91) 11 2793 1342
Pure choriocarcinoma of testis with
rare gingival and skin metastases
metastases, oral metastatic tumours are also uncommon
and comprise only approximately 1% of the malignant
A 22-year-old man with no known significant
comorbidities, was referred by a dermatologist, with
chief complaints of nodular skin swellings over the
sternal region (Fig. 1) and over the left chin area of
ten days, duration, and which were adherent to the
underlying structures. Another swelling over the left
lower gingiva was also present. Fine-needle aspiration
cytology (FNAC) was done from the sternal lesion by
the dermatologist, who was suspicious of malignancy.
Clinical examination revealed a dark-pigmented, non-
tender, nodular swelling, 3 cm in size, over the sternal
region, and another on the chin that was 1 cm in size,
and another fleshy non-pigmented soft tissue swelling
over the left lower gingiva.
Fig. 1 Photograph shows a fleshy hyperpigmented nodular
lesion measuring 3.5 cm over the sternal region.
Singapore Med J 2007; 48(3) : e78
On further questioning, he revealed the presence
of a painless left testicular enlargement, which had been
present for the past two months. FNAC from all the
skin nodules (Figs. 2 & 3) was done, due to non-
availability of the outside FNAC slides for our review.
Histopathological examination was consistent with
the diagnosis of metastatic germ cell tumour. Serum
beta-human chorionic gonadotrophin (HCG) level
was 468 IU/ml (normal range, 0–25 IU/ml). Serum
alpha-foetal protein was 0.426 IU/ml (normal
< 7.2 IU/ml). Serum lactate dehydrogenase was
438 U/L (normal range, 225–450 U/L). Computed
tomography of the chest showed multiple nodular
lesions in both lungs, the largest being 2.3 cm × 1.6 cm
and 1.9 cm × 1.5 cm in the left and right lungs,
respectively. Haematological and biochemical
parameters were unremarkable.
He underwent left high orchidectomy and
histopathological examination showed choriocarcinoma
of the left testis (Figs. 4 & 5). Immunohistochemistry
(IHC) was done after discussion with our tumour board,
and the result was consistent with the diagnosis of
Fig. 6 IHC shows immunostaining with beta-HCG of
syncytiotrophoblasts (Clone 2B1.3, Isotype IgG2a, ×40).
Fig. 2 FNAC of the skin lesions shows cells with a variable
amount of cytoplasm (Pap stain, ×40).
Fig. 3 FNAC of the skin lesions shows cells with large cytoplasm,
pleomorphic nuclei along with multinucleated tumour giant cells
(Pap stain, ×40).
Fig. 4 Photomicrograph of the high orchidectomy specimen
shows choriocarcinoma with syncytiotrophoblasts (bold
arrow) and cytotrophoblasts (thin arrow) (Haematoxylin &
Fig. 5 Photomicrograph of the high orchidectomy specimen
shows choriocarcinoma with syncytiotrophoblasts (bold
arrow) and cytotrophoblasts (thin arrow) (Haematoxylin
& eosin, ×100).
Singapore Med J 2007; 48(3) : e79
pure choriocarcinoma (Fig. 6). He was started on
chemotherapy with the Bleomycin, Etoposide and
Cisplatin (BEP)-based protocol. After completion
of three cycles of chemotherapy, his skin and gingival
nodules regressed completely. There was decline in
the level of serum beta-HCG after all the three cycles
(468⁄200⁄180⁄160 IU/ml). Although the decline
in the levels of beta-HCG was not appropriate, he
was continued on the chemotherapy with the BEP
protocol in view of good clinical response, as
indicated by regression of the skin as well as the
There was no evidence of clinical relapse after
completion of four cycles of chemotherapy. However,
after the fourth cycle of chemotherapy, serum HCG
level rose to 1,000 IU/ml. In view of the rising
beta-HCG, he was planned for brain imaging, with
the suspicion of possible central nervous system
involvement, although he had no neurological
signs and symptoms. He was also planned for salvage
chemotherapy in view of rising beta-HCG levels.
After the fourth cycle of chemotherapy, the patient
was counselled regarding the brain imaging and
change of chemotherapy plan. But he moved to
another centre for treatment and hence, was lost to
Choriocarcinoma is a malignant growth of
trophoblastic cells, which is characterised by
the secretion of HCGs.(5) It usually arises as
gestational choriocarcinoma, from foetal trophoblasts
of a previous hydatidiform mole pregnancy. Rarely,
it arises from germ cells in the testis or ovary.
Choriocarcinoma is conventionally classified as
pure choriocarcinoma, which is composed of only
syncytiotrophoblastic and cytotrophoblastic components,
and mixed germ cell tumour, which contains
choriocarcinoma as one of the components. In males,
it usually occurs as a component of testicular mixed
germ cell tumours, whereas its pure form represents
less than 3% of all the cases.(3) Non-seminomatous
germ cell tumours of the testis usually metastasise
to the retroperitoneal lymph nodes, lung, liver and
brain, but metastases to skin and oral mucosa are
The skin is an uncommon site of metastatic
disease.(8) The overall incidence of metastasis to the skin
is only 0.7%–9%.(1) The most frequently observed
primary sites leading to skin metastases are the breast,
colon and melanoma in women, and the lung, colon
and melanoma in men.(1) With both the genders
combined, colorectal carcinoma is the most common
visceral malignancy leading to skin metastasis.(9)
The anterior chest wall is the most common site
for metastatic skin lesions, followed by the face
and the lower extremities.(9,10) Approximately, 3%–
15% of the patients with skin metastasis have
multiple metastatic sites.(6,7) In general, metastatic
carcinoma of skin is considered a poor prognostic
Similiarly, oral metastatic tumours are also
uncommon and comprise only 1% of the malignant
oral lesions. The most common primary sources of
metastases to the oral region are the breast, lung,
kidney, bone and colon. The breast and lungs are
the most common primary sites for metastasis to the
jawbones and oral soft tissue, respectively. Also,
the most common source of metastases to the oral
cavity is the breast in women, and lungs in men. Renal
cell carcinoma is another common tumour that
metastasise to the oral mucosa.(12) In the oral soft
tissue, the attached gingiva is the most common
site of metastasis, followed by the tongue.(4) The
early manifestations of the gingival metastases may
resemble hyperplastic or reactive lesions, such as
pyogenic granuloma, peripheral giant cell granuloma,
or fibrous epulis.(4,12,13)
Hirshberg et al analysed 157 cases of well-
documented metastatic lesions to the oral cavity.
According to their analysis, the most common oral
site of involvement was the gingiva, followed by the
tongue, tonsil, palate, lip, buccal mucosa and floor
of mouth, in that order.(12) Gingival lesions were
almost equally distributed between the maxilla and
mandible.(12) In dentulous patients, 79% exhibited
metastases to the attached gingiva, whereas in
edentulous patients metastatic lesions were equally
distributed between the tongue and the alveolar
This case is unique in being pure choriocarcinoma
of testis, which accounts for less than 3% of the
testicular neoplasms with rare skin, and even rarer,
oral soft tissue metastases. This also emphasises that
metastases to these sites should be investigated for
the presence of testicular malignancy in men. Thus,
suspicious gingival and skin lesions should be
investigated thoroughly by the dentist and dermatologist,
respectively, for the presence of an underlying
malignancy. Also, the levels of beta-HCG may provide
an important clue to the response of the patient.
As in our case, the decline in levels of beta-HCG
was not appropriate, although clinical response of
the patient was encouraging. Apart from refractory
disease which can lead to raised levels of beta-HCG,
an occult central nervous system should be
kept in mind and brain imaging should be done
Singapore Med J 2007; 48(3) : e80
1. Spencer PS, Helm TN. Skin metastases in cancer patients. Cutis
2. Schwartz RA. Cutaneous metastatic disease. J Am Acad Dermatol
1995; 33:161-82. Comment in: J Am Acad Dermatol 1996; 34:1093.
3. Shimizu S, Nagata Y, Han-yaku H. Metastatic testicular
choriocarcinoma of the skin. Report and review of the literature.
Am J Dermatopathol 1996; 18:633-6.
4. Hirshberg A, Buchner A. Metastatic tumors to the oral region.
An overview. Eur J Cancer B Oral Oncol 1995; 31B:355-60.
5. Robey EL, Schellhammer PF. Four cases of metastases to the penis
and a review of the literature. J Urol 1984; 132:992-4.
6. Gates O. Cutaneous metastasis of malignant disease. Am J Cancer
7. Brownstein MH, Helwig EB. Metastatic tumors of the skin. Cancer
8. Chhieng DC, Jennings TA, Slominski A, Mihm MC Jr. Choriocarcinoma
presenting as a cutaneous metastasis. J Cutan Pathol 1995; 22:374-7.
9. Brownstein MH, Helwig EB. Patterns of cutaneous metastasis.
Arch Dermatol 1972; 105:862-8.
10. Tharakaram S. Metastases to the skin. Int J Dermatol 1988; 27:240-2.
11. Reingold IM. Cutaneous metastases from internal carcinoma. Cancer
12. Hirshberg A, Leibovich P, Buchner A. Metastases to the oral
mucosa: analysis of 157 cases. J Oral Pathol Med 1993; 22:385-90.
13. Rim JH, Moon SE, Chang MS, Kim JA. Metastatic hepatocellular
carcinoma of gingiva mimicking pyogenic granuloma. J Am
Acad Dermatol 2003; 49:342-3.