CASE REPORT – OPEN ACCESS International Journal of Surgery Case Reports 4 (2013) 849–851
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A case of solitary pulmonary metastasis of choroidal melanoma with an exceptionally long disease-free period Teruya Komatsu ∗ , Terumasa Sowa, Koji Takahashi, Takuji Fujinaga Department of General Thoracic Surgery, Nagara Medical Center, Gifu, Japan
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Article history: Received 24 March 2013 Received in revised form 2 June 2013 Accepted 10 June 2013 Available online 16 July 2013 Keywords: Choroidal melanoma Metastases Solitary pulmonary metastasis VATS (video-assisted thoracoscopic surgery)
a b s t r a c t INTRODUCTION: Choroidal melanoma is the most common primary malignant intraocular tumor in adults. And its metastatic spread has been considered to be hematogenous with the liver being the most common site, and a solitary pulmonary metastasis without hepatic involvement is quite rare. PRESENTATION OF CASE: We report the case of a 61-year-old woman with a solitary pulmonary metastasis from choroidal melanoma, which had been originally treated with enucleation 13 years ago. Videoassisted thoracoscopic surgery (VATS) right upper lobectomy was performed for the right upper lobe tumor, which was histopathologically confirmed to be metastatic choroidal melanoma. DISCUSSION: Our case is quite unusual in 2 points; firstly, a solitary pulmonary metastasis without hepatic involvement is rare. Secondly, this mode of metastasis with an exceptionally long disease-free period (13 years) is also rare. CONCLUSION: A pulmonary metastasis from choroidal melanoma is described with reference to relevant literature. © 2013 The Authors. Published by Elsevier Ltd on behalf of Surgical Associates Ltd. Open access under CC BY-NC-ND license.
1. Introduction Melanoma of the choroid (which is part of the uvea) is an uncommon malignancy with a reported incidence of about 6 per million population per year.1 Its diagnosis can be done non-invasively with indirect ophthalmoscopy (IO), A- and B-ultrasonography scans, fundus fluorescein angiography (FFA), and transillumination. Invasive studies such as fine needle aspiration are not routinely recommended due to its significant complications like seeding of tumor cells in the needle track.2 Because of higher blood flow of the choroid, the mode of metastatic spread of choroidal melanoma has been considered to be hematogenous with the liver being the most common site.3 A solitary metastasis without hepatic involvement is quite rare. 2. Presentation of case
right upper lobe. The histopathological examination of the enucleated left eye showed a mixed-cell-type choroidal melanoma without intrascleral or vascular involvement. The size of the melanoma was 10 mm in thickness and 13 mm in diameter. Since then, the patient had been followed up regularly by an ophthalmologist. An abnormal shadow, which was incidentally found on chest radiograph for a health screening, was confirmed to be a solitary nodule measuring 25 mm in diameter in the right upper lobe by computed tomography (CT) scan. F-18fluoro-2-deoxyglucose positron emission tomography/computed tomography (FDG-PET/CT) scan demonstrated a solitary pulmonary nodule with increased FDG uptake. For a diagnostic and therapeutic purpose, video-assisted thoracoscopic surgery (VATS) right upper lobectomy was performed with uneventful postoperative recovery. Intraoperative gross examination revealed a black tumor in the right upper lobe in the vicinity of the pulmonary hilum. Histopathological examination of the tumor resected from the right upper lobe revealed metastatic melanoma (Fig. 1).
A 61-year-old woman who had undergone left ocular enucleation for choroid melanoma 13 years ago was referred to our department for the evaluation of a solitary pulmonary tumor in the
3. Discussion
∗ Corresponding author at: Department of General Thoracic Surgery, Nagara Medical Centre, 1300-7 Nagara, Gifu City, Gifu Prefecture 502-8558, Japan. Tel.: +81 58 232 7755; fax: +81 58 295 0077. E-mail address:
[email protected] (T. Komatsu).
Choroidal melanoma is the most common primary intraocular malignancy in adults. In the general population, it is uncommon with an incidence of 5.3–10.9 cases per million population per year; both sexes are equally affected.1 A small choroidal melanoma (longitudinal diameter 1–3 mm, anterior-posterior diameter ≥5 mm) in the posterior fundus is amenable to several treatment options, including laser photocoagulation, photodynamic therapy, plaque radiation therapy,
2210-2612 © 2013 The Authors. Published by Elsevier Ltd on behalf of Surgical Associates Ltd. Open access under CC BY-NC-ND license. http://dx.doi.org/10.1016/j.ijscr.2013.06.017
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T. Komatsu et al. / International Journal of Surgery Case Reports 4 (2013) 849–851
Fig. 1. (A) Infused computed tomography (CT) scan of the chest showing a moderately enhanced solitary pulmonary tumor in the right upper lobe. (B) The tumor showing positive F-18-fluoro-2-deoxyglucose (FDG) uptake on positron emission tomography (PET) scan. (C) Black tumor around the pulmonary hilum. (Arrow head showing the superior pulmonary vein encircled with a rubber tape.). (D) Histopathological section of the resected specimen demonstrating metastatic choroidal melanoma containing melanin pigment. (Hematoxylin and eosin; ×200).
external beam charged particle radiation therapy, transpupillary thermotherapy, location tumor resection, and enucleation. Enucleation is the traditional method of treating medium (longitudinal diameter 2.5–10 mm, anterior-posterior diameter ≤16 mm) and large (longitudinal diameter ≥10 mm; anterior-posterior diameter ≥16 mm) choroidal melanoma. Irrespective of the availability of alternative treatment modalities, e.g., plaque radiotherapy, proton beam radiotherapy, and trans-pupillary thermotherapy, the survival rates of patients with choroidal melanoma have not changed in the past 30 years.1 Recently gamma knife surgery has been also studied for its potential, however, its role in treatment has yet to be elucidated.2 Considering that the success of choroidal melanoma depends on vision-related quality of life as well as survival and that a variety of treatment modalities are available, its therapeutic strategy should be individualized.2 The cumulative rates of metastases in the Collaborative Ocular Melanoma Study (COMS) at 5 and 10 years after treatment were 25% and 34%, respectively. Common sites of metastases include liver (90%), lung (24%), and bone (16%).3,4 The report by Lorigan et al. describes that isolated pulmonary metastases from ocular melanoma are very rare, accounting for 3.6% of metastatic ocular melanoma because extra-ocular spread to other organs has almost always been seen in association with hepatic metastases.5 Patients with metastases confined to extrahepatic locations have longer survival (19 versus 7 months).6 Aoyama et al. reported longer survival after complete surgical removal of metastatic choroidal melanoma in selected patients, including 2 with a solitary lung nodule.7 There have been reports regarding predictive factors of the metastatic potential of choroidal melanoma. According to the COMS classification, primary tumor size is one of the best parameters to predict the risk of metastatic disease. Diener-West et al. have summarized that the 5-year survival rates after enucleation were 84% for small, 68% for medium-sized, and 47% for large tumors.8 In our case, the size of the original choroidal melanoma was classified as large. In terms of TNM cancer classification by American joint committee on cancer, our case was staged as T3aN0M0, stage B with the 10-year survival rate of 68%.9 Therefore, considering the higher recurrence rate, it is quite reasonable that the patient had
seen an ophthalmologist and underwent chest radiography annually, even though more than 10 years had passed since the primary ocular lesion was treated. From our experience, patients treated for choroidal melanoma should be followed up for more than 10 years. So far, there has been no universally accepted modality of screening for metastatic choroidal melanoma. In the COMS trial, chest radiography and liver function tests were performed every 6 months for at least 5 years.3 Klingenstein et al. reported that wholebody FDG-PET/CT imaging is a sensitive modality in the follow-up of choroidal melanoma patients though the amount of radiation exposure from the FDG-PET/CT imaging should be worried about.10 Considering our case in which solitary pulmonary metastasis was FDG-avid on PET scan, FDG-PET/CT imaging might be a sensitive modality for detecting metastatic choroidal melanoma. In terms of screening, the effectiveness of CT scan for metastatic choroidal melanoma has not been elucidated yet. 4. Conclusion Choroidal melanoma has the potential to metastasize even after more than 10 years after treatment of the primary ocular lesion. Regular long-term follow-up for patients with metastatic choroidal melanoma is mandatory. If the metastatic lesion is extrahepatic and resectable, surgery should be the treatment of choice. Conflict of interest The authors declare that they have no competing interests. Funding None. Ethical approval Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy
CASE REPORT – OPEN ACCESS T. Komatsu et al. / International Journal of Surgery Case Reports 4 (2013) 849–851
of the written consent is available for review by the Editor-in-Chief of this journal on request. Author contributions Teruya Komatsu, MD; Study design, data collections, data analysis, and writing. Terumasa Sowa, MD and Koji Takahashi, MD; Study design and data analysis. Takuji Fujinaga, MD; writing and supervision. Acknowledgements We thank Mr. Keiji Yata, Department of Clinical Laboratory at the Nagara Medical Center, for providing a photomicrograph of the specimen demonstrating metastatic choroidal melanoma. References 1. Papastefanou VP, Cohen VM. Uveal melanoma. Journal of Skin Cancer 2011;2011(2011):573974.
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2. Singh P, Singh A. Choroidal melanoma. Oman Journal of Ophthalmology 2012;5(1):3–9. 3. Diener-West M, Reynolds SM, Agugliaro DJ, et al. Screening for metastasis from choroidal melanoma: the collaborative ocular melanoma study group report 23. Journal of Clinical Oncology 2004;22(12):2438–44. 4. Group COMS. Assessment of metastatic disease status at death in 435 patients with large choroidal melanoma in the collaborative ocular melanoma study (COMS): COMS report no. 15. Archives of Ophthalmology 2001;119(5):670–6. 5. Lorigan JG, Wallace S, Mavligit GM. The prevalence and location of metastases from ocular melanoma: imaging study in 110 patients. American Journal of Roentgenology 1991;157(6):1279–81. 6. Saraiva VS, Rodriguez-Reyes AA, Chen MF, et al. Metastatic choroidal melanoma presenting as a solitary pulmonary nodule. Canadian Journal of Ophthalmology 2005;40(1):72–4. 7. Aoyama T, Mastrangelo MJ, Berd D, et al. Protracted survival after resection of metastatic uveal melanoma. Cancer 2000;89(7):1561–8. 8. Diener-West M, Hawkins BS, Markowitz JA, et al. A review of mortality from choroidal melanoma. II. A meta-analysis of 5-year mortality rates following enucleation, 1966 through 1988. Archives of Ophthalmology 1992;110(2):245–50. 9. American joint committee on cancer. Cancer staging manual. 7th ed. Springer; 2010. 10. Klingenstein A, Haug AR, Nentwich MM, et al. Whole-body F-18-fluoro-2deoxyglucose positron emission tomography/computed tomography imaging in the follow-up of metastatic uveal melanoma. Melanoma Research 2010;20(6):511–6.
