Yang et al.

World Journal of Surgical Oncology (2015) 13:326

DOI 10.1186/s12957-015-0741-0

CASE REPORT Open Access

Local anesthetic thoracoscopy for the

diagnosis of metastatic pleural melanoma
originated from oral malignant melanoma:
case report and comments
Bingjun Yang1, Qingzhao Li2, Hui Zhao1, Huibin Liu3, Tao Tang4 and Chunyang Jiang1*

Abstract
Background: Oral malignant melanoma (OMM) is an aggressive tumor with very low survival rate and easy to
metastasize. Pleural metastatic melanoma via primary OMM is rare.
Case presentation: In this report, we presented a case of metastatic malignant melanoma of the pleura originated
from OMM. A 54-year-old man without primary skin lesion was diagnosed multiple nodular shadows, pleural invasion,
and pleural effusion by chest computed tomography (CT). One cyst-form tumor on the tongue base was observed by
bronchoscopy, which was diagnosed as OMM by pathological examination and then was resected. After getting the
tumor tissues from the pleura by pleural biopsy surgery, the diagnosis of pathological examination was pleural
metastatic melanoma. Furthermore, tumor cells displayed a positive immunoreaction for melanocytic markers
S100 and HMB-45 combining with positive vimentin and cytokeratin AE1/AE3. The patient was therefore
diagnosed with metastatic melanoma of the left pleura and the primary melanoma was OMM.
Conclusions: According to this case, we could draw the conclusion that pleural metastasis from OMM was very rare
and thoracoscopy preceded under local anesthesia is an important method for its accurate diagnosis.
Keywords: Pleural melanoma, Thoracoscopy, Oral malignant melanoma, Local anesthesia, Diagnosis

Background Surgery can be performed as an effective treatment at the

Melanomas, including two subtypes, are originated from early phase of the tumor. Unfortunately, melanoma is
melanoblasts or melanocytes which can have a benign or prone to metastasis by lymphatic pathway to regional
malignant clinical course. They mainly occur on the skin lymph nodes and even by circulation to distant sites,
and are relevant to ultraviolet light exposure as common which can significantly worsen the prognosis [5, 6]. The
tumors in human [1]. The incidence of melanoma is in- median survival time for metastatic melanoma patients is
creasing about 5 % per year worldwide [2]. Malignant only about 69 months [7, 8].
melanoma (MM) is the most aggressive skin cancer ori- Primary melanoma of the thorax, which mainly involves
ginating from melanocytes with a high degree of pheno- the lung and pleura, is exceptionally rare. In this report, we
typic plasticity, which is primarily located in the skin but presented a case that was diagnosed as metastatic MM of
also found in eyes, ears, mouth, gastrointestinal tract, the pleura by pleural biopsy surgery, which was originated
genital mucosa, and leptomeninges [3]. Although melan- from an oral malignant melanoma (OMM). The initial
oma accounts for only 5 % of all malignant skin tumors, it symptoms of this patient were chest pain and dyspnea for
has occupied 75 % deaths of all skin tumor patients [4]. 2 months. Chest computed tomography (CT) which was
performed on a 64-row multidetector CT scanner
* Correspondence: [email protected]

(Somatom Sensation, Siemens, Erlangen, Germany) showed

Equal contributors
1
Department of Thoracic Surgery, Tianjin Union Medicine Centre, 190 Jieyuan
multiple nodular shadows, pleural invasion, and pleural
Road, Hongqiao District, Tianjin 300121, Peoples Republic of China effusion, and at first, the case was misdiagnosed as lung
Full list of author information is available at the end of the article

2015 Yang et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Yang et al. World Journal of Surgical Oncology (2015) 13:326 Page 2 of 6

Fig. 1 The chest CT imaging of the pleural effusion and lesions. Before operation, pleural effusion and lesions in the left thorax and multiple
metastases in the right lung can be seen in CT scan imaging (a and b). After complete drainage of pleural effusion, metastatic neoplasms in
pleura, enlargement of mediastinal lymph nodes, and lung metastases were clearly emerged by countercheck CT scan (c and d). The pleural
lesions were pointed by blue arrows in d. a and c Lung window. b and d Mediastinum window

Fig. 2 NBI bronchoscopy. One cystoma was on the left side of the tongue base (a). The cystoma was observed by bronchoscopy and pointed by
black arrows. Abnormalities were not seen in the glottis and tracheal juga (b and c). Bronchial stenosis of left pulmonary segments can be seen in d
Yang et al. World Journal of Surgical Oncology (2015) 13:326 Page 3 of 6

Fig. 3 The pathological results of the resected tumor from the tongue base and pleural tumor tissue and immunohistochemical detection of the
pleural tumor tissue. The tissues histopathology changes were observed in light microscope (Nikon Eclipse 80i, Tokyo, Japan) and photos were taken.
Postoperative pathological results for the tongue base and pleural tumor tissue, respectively (a and b) (200). The immunohistochemical results of the
pleural tumor tissue are shown in cf, which are S100, HMB-45, vimentin, and cytokeratin AE1/AE3, respectively, and the magnification are all of 100

and pleural metastatic carcinoma. Therefore, thoracoscopy, CT scan (Fig. 1a, b). The previous medical history reported
including video-assisted thoracoscopic surgery (VATS) and no primary skin lesion including melanin stain. The re-
simple rigid thoracoscopy, is the key to the accurate diag- lated examines were finished before thoracoscopy pleural
nosis of this disease. biopsy operation. One cyst of the neoplasm on the left side
of the tongue base and multiple bronchial stenosis of left
Case presentation pulmonary segments were observed by narrow band im-
The patient was a 54-year-old Han Chinese man who, in aging (NBI) bronchoscopy (Olympus, EVIS LUCERA)
June 2013, was diagnosed as double-sided pleural effusion (Fig. 2). The tumor was then resected and diagnosed as
which was combined with the left multifocal pleural OMM by pathological examination (Fig. 3a) and the
lesions and multiple lung metastases in the right lung by immunohistochemical staining results (Additional file 1:

Fig. 4 The semi-rigid thoracoscopy was used in pleural biopsy surgery

Yang et al. World Journal of Surgical Oncology (2015) 13:326 Page 4 of 6

Figure S1). The results of tuberculosis antibody (TB-Ab) results suggested that the HMB-45, S-100, vimentin, and
and TB-DNA in serum were all negative. Serum tumor AE1/AE3 (antibodies were all purchased from Santa Cruz
markers for lung carcinoma including carcino-embryonic Biotechnology (Santa Cruz, CA)) were all significantly
antigen, carbohydrate antigen 72-4, squamous cell carcin- positive expressions (Fig. 3bf).
oma, cyfra 21-1, cytokeratin 19 fragments, and ferritin After almost complete drainage of pleural effusion, re-
were all in normal range. However, carbohydrate antigen markable multiple metastatic neoplasms in the pleura,
125 and neuron-specific enolase were about three times enlargement of mediastinal lymph nodes, and several
and two times higher than the upper limits, respectively. lung metastases could be clearly observed by counter-
Subsequently, the pleural biopsy surgery of the left check CT scan (Fig. 1c, d). During the inpatient period,
thorax was preceded by semi-rigid thoracoscopy (model detections of brain CT scan, abdominal Doppler ultra-
LTF-240, Olympus, Tokyo, Japan) (Fig. 4) with video as- sound, and so on, there were no evidence to confirm
sistance under local anesthesia using 10 mL 1 % Lidocaine other organs and any other lymphatic node metastases.
injected into the subcutaneous tissue. The thoracoscopy Subsequently, there was no further treatment, and less
was entered from a single port in the sixth intercostal than 6 months of follow-up, the patient died.
space. After extraction of 1500 mL bloody pleural fluid, This case report and related experimental protocols
multiple violet black neoplasms can be seen in the parietal were approved by the ethics committee of Tianjin Union
pleura, which were different sizes, very brittle, and prone Medicine Centre of China.
to bleeding (Fig. 5). After biting from several sections of
these lesions, the tissues were used for pathological de- Discussion
tection. Pleural effusion was continuously discharged In the past several decades, the incidence of melanoma has
by using closed drainage tube. Cytology examination of been steadily rising with an annual increase of 38 %
pleural effusion displayed red blood cells in the field of worldwide [9]. The most common form of melanoma is
vision and severe abnormity of cell nucleus. the cutaneous or the ocular form. As neural crest-derived
Postoperative pathological reports showed that these neo- cells, melanocytes could migrate to the skin, mucous mem-
plasms were pleural metastatic melanoma by hematoxylin- branes, and other sites. Melanoma metastasizing to thorax
eosin (HE) staining, and combined immunohistochemical is common, but primary pulmonary or pleural melanoma

Fig. 5 The main observation of pleural biopsy surgery in the right parietal pleura. Multiple violet-black neoplasms can be seen in the parietal
pleura by thoracoscopy with video assistance, being different sizes, very brittle, and prone to bleeding, and these lesions are pointed by white
arrows (ac). d The tumor tissues were gained by biopsy forceps. The used forceps is pointed by blue arrow
Yang et al. World Journal of Surgical Oncology (2015) 13:326 Page 5 of 6

is extremely rare. Therefore, MM has very seldom been de- examination and pathological and immunohistochemistry
scribed as a primary tumor in the pleura or lower respira- detection. Pleural metastasis via OMM was very rare, and
tory tract, and there are only very limited literatures that thoracoscopy preceded under local anesthesia is an effect-
have been reported [10]. Since metastasis of MM to the ive and safety method for its accurate diagnosis.
lung or pleura is relatively common, it is very important to
distinguish primary from secondary melanomas. Consent
OMM, which was first described by Weber in 1859, is a The patient and his family members were informed and
much rare neoplasm located at the basal layer of the oral have consented for the publication of this report. A copy
mucous membranes owing to the uncontrolled growth of of the written consent is available for review by the
melanocytes [11]. Mucosal melanoma accounts for only Editor-in-Chief of this journal.
0.5 % of all oral tumor involving the sinonasal cavity, oral
cavity, pharynx, larynx, and upper esophagus [12]. Oral
melanomas occur slightly more often in males (2.8:1, male Additional file
to female ratio) and with an average age of 56 years (the
Additional file 1: Figure S1. The immunohistochemical results of the
age range is from 20 to 83 years) [13]. OMM belongs to primary tongue melanoma tissue are shown in (a), (b), (c) and (d), which
head and neck mucosal melanomas (HNMM) and fre- are S100, HMB-45, vimentin, and cytokeratin AE1/AE3, respectively, and
quently exhibits postoperative recurrence and distant the magnification are all of 200 (JPG 2042 kb)
metastasis. For HNMM patients, surgery is recommended
if indicated, and surgery combined with postoperative Abbreviations
radiotherapy is also recommended for dramatically im- CT: computed tomography; HE: hematoxylin-eosin; HNMM: head and neck
mucosal melanoma; MM: malignant melanoma; NBI: narrow band imaging;
proved local control of the tumor bed. Radiotherapy and OMM: oral malignant melanoma; TB-Ab: tuberculosis antibody; VATS: video-
immunological therapy could be potential options for pa- assisted thoracoscopic surgery.
tients without surgery chance [14].
In the present case, the primary site of melanoma was Competing interests
previously found in the root of the tongue without pri- The authors declare that they have no competing interests.
mary skin lesion. After obtaining the tumor tissues from
Authors contributions
several sites of parietal pleura by pleural biopsy surgery, C.Y. J., H.B.L., and B.J.Y. conceived and designed the experiments and drafted the
the diagnosis of pathological examination was pleural manuscript. B.J.Y., H.Z., and C.Y. J. performed the narrow band imaging (NBI)
metastatic melanoma through HE staining method. Fur- bronchoscopy detection, video-assisted thoracoscopic surgery (VATS) and simple
rigid thoracoscopy operations. Y.G. and T.T. performed postoperative pathological
thermore, tumor cells displayed a positive immunoreac- detections and immunohistochemical results including HMB-45, S-100, vimentin,
tion for melanocytic markers (S100 and HMB-45, which and AE1/AE3. C.Y.J. prepared all figures. H.B.L., C.Y.J., and B.J.Y. wrote the paper.
are frequently expressed in primary oral melanomas and All authors discussed the results, reviewed the manuscript, and approved the
final manuscript.
helpful to confirm the diagnosis [15]). In addition, both
vimentin and cytokeratin AE1/AE3 also appeared as Author details
1
positive expressions. We therefore ascertained that the Department of Thoracic Surgery, Tianjin Union Medicine Centre, 190 Jieyuan
Road, Hongqiao District, Tianjin 300121, Peoples Republic of China. 2School
metastatic melanoma of the left pleura was metastasized of Public Health, North China University of Science and Technology,
from OMM. Tangshan 063001, Hebei, Peoples Republic of China. 3Office of Clinical Drug
Modern thoracoscopy provides a potentially less inva- Trial Institution, The Affiliated Tumor Hospital of Xinjiang Medical University,
Urumqi, Xinjiang 830011, Peoples Republic of China. 4Department of
sive means to diagnose and to treat a variety of intratho- Pathology, Tianjin Union Medicine Centre, 190 Jieyuan Road, Hongqiao
racic diseases. Simple rigid thoracoscopy or VATS is safe District, Tianjin 300121, Peoples Republic of China.
and effective for the diagnosis of both benign and malig-
Received: 10 October 2015 Accepted: 23 November 2015
nant pleural disease which has very high sensitivity (80
to 100 %) [16]. For the present patient, the color and the
appearance of the melanoma are easy to recognize under References
thoracoscopy, with the potential to limit complications 1. Miller AJ, Mihm MC. Melanoma. N Engl J Med. 2006;355(1):5165.
and to reduce morbidity and hospital stays for thoracot- 2. Jemal A, Siegel R, Xu J, Ward E. Cancer statistics, 2010. CA Cancer J Clin.
2010;60(5):277300.
omy. Pleural tumorous diseases are one important indi- 3. McCourt C, Dolan O, Gormley G. Malignant melanoma: a pictorial review.
cation for thoracoscopy with video assistance and can Ulster Med J. 2014;83(2):103.
easily be diagnosed accurately. 4. Rigel DS, Russak J, Friedman R. The evolution of melanoma diagnosis:
25 years beyond the ABCDs. CA Cancer J Clin. 2010;60(5):30116.
5. McMeniman E, DeAmbrosis K, DeAmbrosis B. Risk factors in a cohort of
Conclusions patients with multiple primary melanoma. Australas J Dermatol. 2010;
We have presented a very rare case of amelanotic mel- 51(4):2547.
6. De Vita F, Giordano G, Fabozzi A, Guerrera B, Fabozzi T, Fasano M,
anoma of the left parietal pleura that was diagnosed as et al. Metastatic melanoma: an unusual presentation. Tumori.
a metastatic tumor from OMM by detailed physical 2011;97(1):1302.
Yang et al. World Journal of Surgical Oncology (2015) 13:326 Page 6 of 6

7. Garca M, del Muro XG, Tres A, Crespo C, Valladares M, Lopez JJ, et al. Phase
II multicentre study of temozolomide in combination with interferon alpha-
2b in metastatic malignant melanoma. Melanoma Res. 2006;16(4):36570.
8. Tarhini AA, Agarwala SS. Cutaneous melanoma: available therapy for
metastatic disease. Dermatol Ther. 2006;19(1):1925.
9. Pour MSH. Malignant melanoma of the oral cavity. J Dentistry Tehran Univer
Med Sci. 2007;4(1):4451.
10. Ohata Y, Haga T, Ogata S, Nakanishi K, Kawai T. Malignant amelanotic
melanoma of the pleura without primary skin lesion: an autopsy case
report. Acta Med Okayama. 2009;63(6):37984.
11. Gondivkar SM, Indurkar A, Degwekar S, Bhowate R. Primary oral malignant
melanomaa case report and review of the literature. Quintessence Int.
2009;40(1):41-46.
12. Cockerell CJ. The pathology of melanoma. Dermatol Clin. 2012;30(3):44568.
13. Gu GM, Epstein JB, Morton TH. Intraoral melanoma: long-term follow-up
and implication for dental clinicians. A case report and literature review.
Or Surg Or Med Or Pa. 2003;96(4):40413.
14. Li W, Yu Y, Wang H, Yan A, Jiang X. Evaluation of the prognostic impact of
postoperative adjuvant radiotherapy on head and neck mucosal melanoma:
a meta-analysis. BMC Cancer. 2015;15:758.
15. de-Andrade BA, Toral-Rizo VH, Len JE, Contreras E, Carlos R, Delgado-
Azanero W, et al. Primary oral melanoma: a histopathological and
immunohistochemical study of 22 cases of Latin America. Med Oral Patol
Oral Cir Bucal. 2012;17(3):e383388.
16. Soni A, Bansal V, Goel A. The role of thoracoscopy in diagnosis and
treatment of pleural disease. World J Laparoscopic Surg. 2012;5(1):415.

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