Cervical mature teratoma 17 years after initial treatment of testicular teratocarcinoma: Report of a late relapse

Article (PDF Available)inWorld Journal of Surgical Oncology 5(1):1 · February 2007with20 Reads
DOI: 10.1186/1477-7819-5-1 · Source: PubMed
Abstract
Late relapses of testicular germ cell tumor are uncommon. We report a case of cervical mature teratoma appeared 17 years after treatment of testicular teratocarcinoma. A 20-year-old patient underwent left sided orchiectomy followed by systemic therapy and retroperitoneal residual mass resection in 1989. He remained in complete remission for 200 months. In 2005 a huge left supraclavicular neck mass with extension to anterior mediastinum appeared. Radical surgical resection of the mass was performed and pathologic examination revealed mature teratoma. This is one of the longest long-term reported intervals of a mature teratoma after treatment of a testicular nonseminoma germ cell tumor. This case emphasizes the necessity for follow up of testicular cancer throughout the patient's life.
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World Journal of Surgical Oncology
Open Access
Case report
Cervical mature teratoma 17 years after initial treatment of
testicular teratocarcinoma: report of a late relapse
Ramesh Omranipour* and Mina Alavion
Address: Department Of Surgical Oncology, Cancer Institute, Tehran University Of Medical Science, Tehran, Iran
Email: Ramesh Omranipour* - omranipour@sina.tums.ac.ir; Mina Alavion - alavion@sina.tums.ac.ir
* Corresponding author
Abstract
Background: Late relapses of testicular germ cell tumor are uncommon. We report a case of
cervical mature teratoma appeared 17 years after treatment of testicular teratocarcinoma.
Case presentation: A 20- year- old patient underwent left sided orchiectomy followed by
systemic therapy and retroperitoneal residual mass resection in 1989. He remained in complete
remission for 200 months. In 2005 a huge left supraclavicular neck mass with extension to anterior
mediastinum appeared. Radical surgical resection of the mass was performed and pathologic
examination revealed mature teratoma.
Conclusion: This is one of the longest long-term reported intervals of a mature teratoma after
treatment of a testicular nonseminoma germ cell tumor. This case emphasizes the necessity for
follow up of testicular cancer throughout the patient's life.
Background
The prognosis for nonseminomatous germ cell tumor
(NSGCT) of the testis has been dramatically improved by
using a treatment protocol of cisplatin-based chemother-
apy followed by surgical resection of residual tumor mass.
The complete response rate of disseminated germ cell
tumor with this protocol is between 70%–80%.
Most patients who relapse do so within the first year of
therapy. Late relapse is defined as recurrence after a
relapse- free interval of more than two years after comple-
tion of primary treatment [1]. The cumulative risk of late
relapse in patients appearing relapse -free at two years
after first line chemotherapy is 4% in ten years [2].
The incidence of late relapse after cisplatin based chemo-
therapy of germ cell tumor is related to initial tumor bur-
den and patients with bulky retroperitoneal disease
appear to be at an increased risk of late relapse. As tumor
markers do not rise in one quarter of late relapses, they
should undergo CT scans at annual follow-up evaluations.
In the remaining patients, history, physical examination,
tumor markers and chest X-ray may allow to detect the
majority of late asymptomatic relapses [2].
Herein we report a case of late relapse of a bulky teratocar-
cinoma, 17 years after completion of treatment as a
mature teratoma of the neck and upper thorax.
Case presentation
A 36 year-old man was admitted to our hospital with chief
complaint of left sided neck mass. He had a history of left
orchiectomy in 1988 in another center because of testicu-
lar teratocarcinoma. At that time he was referred to our
center because of huge retroperitoneal mass medial to the
left kidney (Figure 1). The first line chemotherapy with
Published: 04 January 2007
World Journal of Surgical Oncology 2007, 5:1 doi:10.1186/1477-7819-5-1
Received: 14 July 2006
Accepted: 04 January 2007
This article is available from: http://www.wjso.com/content/5/1/1
© 2007 Omranipour and Alavion; licensee BioMed Central Ltd.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0
),
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
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bleomycin and etoposide and cisplatin followed by retro-
peritoneal mass resection revealed mature teratoma and
lead to complete remission. He had been in long-term
remission for 17 years, but he had regular follow-up for
the first 5 years. In November 2005, he was admitted
again because of 5 × 5 cm left supraclavicular mass, which
had appeared the year before. In physical exam there was
no other abnormality and pathologic sign. Serum markers
(AFP, beta HCG and LDH) were normal. CT scan of the
thorax showed a 5 × 5 × 8 cm lobulated neck mass with
extension to anterior mediastinum. (figure 2). The other
parts especially paraaortic area were normal in imaging
studies. Incisional biopsy of the neck mass confirmed
mature teratoma so it was treated by radical surgical resec-
tion (figure 3). The specimen consists only of teratoma
without any other components especially immature ter-
atoma.
Discussion
Germ cell tumours, which are in complete remission two
years after treatments have a high probability of cure and
reports of late relapse, are rare (1.3%–6.2%) [2-6]. Late
relapse may occur at any time; of 81 patients treated for
late relapse at Indian university, 47 patients (60%)
relapsed more than 5 years after the achievement of a
complete initial response [7].
The possible mechanisms of development of a late relapse
in germ cell tumors include the followings: malignant
degeneration of mature teratoma to germinal malignancy,
growth of an occult testicular tumor not eliminated by
chemotherapy, development of a second primary germi-
nal malignancy, persistent microscopic viable tumor with
an atypical less aggressive biologic behaviour [8].
Computed tomography scan showing retroperitoneal residual mass after systemic chemotherapy in 1989Figure 1
Computed tomography scan showing retroperitoneal residual mass after systemic chemotherapy in 1989.
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Patient with bulky retroperitoneal disease and patient
found to have teratoma following cisplatin-based chemo-
therapy appear to be at an increased risk of late relapse. Of
51 patients with mature teratoma in resected retroperito-
neal residual tumor masses after chemotherapy, 9 patients
(17.6%) relapsed. In five patients (9.8 %) relapse resulted
from growing mature teratoma [9].
Teratoma is a chemo resistant, nonseminomatous germ
cell tumor composed of somatic cell type from two or
more germ layers and is derived from a toti potential,
malignant precursor cell (embryonal carcinoma or yolk
sac tumor). Although teratoma is a benign tumor but its
biologic potential is unpredictable and it should be
resected completely because it may grow and become
unresectable.
Disease free survival following resection of teratoma is
related to completeness of resection; therefore, there are
significant advantages to surgery with low volume disease.
Moreover, there is the risk of malignant transformation of
teratoma to carcinoma or sarcoma [10], so unresected ter-
atoma may result in late relapse. A late relapse often
shows a slow growth and usually responds to chemother-
apy poorly. Complete surgical resection of late relapse is
preferred mode of therapy but cure rate are relatively low
in patients with viable cancer. In addition, patients with
symptomatic disease and patients presenting with visceral
metastases carried a poor prognosis [2].
Herein we report a case of late relapse in the left side of
neck 17 years after treatment of left testicular teratocarci-
noma. The incidence of cervical metastasis in testicular
germ cell tumor is about 5% [11]. In germ cell tumour,
metastatic disease first involves the retroperitoneal lymph
nodes, then the tumor spreads via thoracic duct to the lat-
ter's termination near the junction of the left internal jug-
ular and subclavian veins, the area where we resected the
mature teratoma of our patient. The mechanisms of late
relapse in the neck and upper thorax may include the
altered lymphatic drainage from an incompletely resected
spermatic cord, a second primary extragonadal tumor
Computed tomography scan showing cervical mass with extension to anterior mediastinum in 2005Figure 2
Computed tomography scan showing cervical mass with extension to anterior mediastinum in 2005.
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focus, or growing of a mature teratoma. Late relapse may
occur at any time. In a recent study from Memorial Sloan
Kettering Cancer Center the medium time to relapse for
17 patients among 551 patients who had previously com-
plete response to first line chemotherapy was 7.8 years [5].
In a recent analysis of 122 cases of late relapse medium
time to relapse was 64.5 months in nonseminomatous
germ cell tumours[12]. Lehman et al. reported a case of
retroperitoneal mature teratoma 15 years after initial
treatment of testicular germ cell tumor [13]. Late relapse
was observed up to 32 years after initial treatment [7].
Whereas chemotherapy has only minor curative potential
in the treatment of late relapse, patient with localized
resectable disease can be cured. Modified neck dissection
has a demonstrated valuable role in the treatment of met-
astatic non-seminomatus germ cell tumours [14].
Our patient has been well with no evidence of disease
after resection of cervical mature teratoma until the date
of last follow-up (October 2006). However, continued
close follow-up of this case is necessary because the large
tumor burden of teratoma is a significant adverse factor
predictive for further relapse [15]. In our opinion, a rea-
sonable follow up schedule would evaluate the patient
monthly in the first year. The frequency of visits should be
decreased to 2–3 months in the second and third year,
and every 6 months in year 4 and 5, and annually thereaf-
ter. Physical examination, tumor markers and radiological
examinations are complementary. Annual CT scan can
detect late relapse at an asymptomatic phase.
Conclusion
This case emphasizes the necessity of annual follow-up
evaluation of testicular germ cell cancer patient through
Radical resection of cervical mature teratomaFigure 3
Radical resection of cervical mature teratoma.
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their life, in order to detect the majority of late relapse at
an asymptomatic phase.
Competing interests
The author(s) declare that they have no competing inter-
est.
Authors' contributions
RO carried out the surgery and drafted the manuscript.
MA assisted in operation and participated in drafting the
manuscript.
All authors read and approved the final manuscript.
Acknowledgements
Written consent was obtained from the patient for publication of his
records and photographs.
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    • "A Medline search was made on all publications involving cervical teratomas occurring in adults yielded less than 50 such case reports. However, if we exclude those occurring in thyroid, base of skull and along the cervical spine, there were only 10 cases reported (Table 1) [2,5678910111213. The aetiology of the cervical neck teratomas remains unknown. "
    [Show abstract] [Hide abstract] ABSTRACT: Cervical neck teratoma in adult is rare. It has a distinctly different clinicopathological behavior compared to those of the neonate and infant. We report an 18-year-old man who presented with a benign cervical neck teratoma, and present a review of cervical neck teratoma in the literature. Surgery is the primary modality of treatment as malignant transformation occurs during adulthood. The diagnosis of malignancy is based on histopathologic examination. Adjuvant chemotherapy is indicated when malignancy is confirmed.
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    • "This is a necessary step since 50% of men with retroperitoneal tumors also have testicular carcinoma in situ, a precursor for testicular germ cell tumors [5]. Long-term care also involves advising patient to follow up with annual CT to detect relapse at an asymptomatic phase [8]. "
    [Show abstract] [Hide abstract] ABSTRACT: Teratomas are bizarre neoplasms derived from embryonic tissues that are typically found only in the gonadal and sacrococcygeal regions of adults. Retroperitoneal teratomas are rare and present challenging management options. We report here the case of a histologically unusual retroperitoneal tumor detected on computed tomography during the workup of abdominal pain in a 32-year-old male. The evaluation and treatment of this condition and a review of the literature are included in this paper.
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