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Show Unilateral Internuclear Ophthalmoplegia as an Isolated Presentation of Metastatic Melanoma Sunil Kumar, MS, FRCS, Shefalee S. Kent, MD, FRCSC, Arun N. Sundaram, MD, MSc, FRCPC, Manas Sharma, MD Abstract: Multiple sclerosis in younger patients and brain-stem infarction in the elderly are the most common causes of internuclear ophthalmoplegia (INO). We report unilateral INO as the isolated clinical manifestation of a large, solitary, metastatic melanoma in the pons. Brain metasta-sis can present as INO. Journal of Neuro-Ophthalmology 2015;35:54-56 doi: 10.1097/WNO.0000000000000171 © 2014 by North American Neuro-Ophthalmology Society Brainstem tumors can cause various gaze disorders (1,2). Melanoma is the second most common cause of brain metastasis after carcinoma of lung (3) and has a highly characteristic signal pattern on magnetic resonance imaging (MRI). We report INO as the presenting manifestation of solitary metastatic melanoma. The metastasis was remark-able in its large size and the absence of other neurologic findings. CASE REPORT A 52-year-old man was admitted to hospital with a com-plaint of horizontal diplopia for 1 day. His medical history was remarkable for Crohn disease, amyloidosis, and intes-tinal melanoma. He had undergone bowel resection on 3 occasions for Crohn disease. Amyloidosis caused renal failure leading to a renal transplant 10 years earlier. Intestinal melanoma was detected 1 year before admission, and he received chemotherapy for 5 months. He developed metastases in the pelvis and both femurs and was treated with radiotherapy. Neuroophthalmic examination revealed visual acuity of 20/30, right eye and 20/25, left eye. Color vision testing, pupillary reactions, visual fields, and fundi were normal. The patient was unable to adduct the left eye using saccades, pursuit, or vestibular-ocular reflex motion, but he was able to converge. Ductions were full in right eye. Abducting jerk nystagmus of the right eye was present in right gaze. Saccades were normal in the right eye but rightward saccades in the left eye from abduction to the midposition were slow. Vertical eye movements were intact, and there was no vertical ocular misalignment. The remainder of the neurological exam-ination was normal. Brain MRI demonstrated a solitary 1.7 cm contrast-enhancing mass localized within the pons with partial effacement of fourth ventricle (Fig. 1). Results of susceptibility-weighted imaging were consistent with the presence of blood products within the tumor (Fig. 2). These MRI findings were characteristic of metastatic melanoma. Multiple focal abnormalities were also noted in the cranial vault consistent with metastatic disease. The pontine metastasis was treated with gamma knife radiosurgery followed by whole brain radiation and chemo-therapy with fotemustine. Four months later, there was marked improvement in the pontine metastasis (Fig. 3). At 5- and 9-month follow-up examinations, the range of adduction of left eye was full and jerk nystagmus of the abducting right eye had resolved. Slow saccades of the ad-ducting left eye were the only residual finding of the pre-viously complete INO. DISCUSSION Approximately 5% of reported cases of INO are due to neoplastic cause (2,4), which include meduloblastoma (5), glioma (6), cavernoma (7), astrocytoma (5), dermoid and Division of Neurology, and Department of Ophthalmology and Vision Sciences (SK, SS, ANS), University Health Network, University of Toronto, Toronto, Ontario, Canada; and Division of Neuroradi-ology, Department of Medical Imaging (MS), University Health Network, University of Toronto, Toronto, Ontario, Canada. The authors report no conflicts of interest. Address correspondence to Sunil Kumar, FRCS, MS, Eye Specialist Hospital, PO Box 39455, Dhahran 31942, Saudi Arabia; E-mail: sunkaru79@hotmail.com 54 Kumar et al: J Neuro-Ophthalmol 2015; 35: 54-56 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. epidermoid tumor (8), carcinomatous meningitis (9), and paraneoplastic syndrome (10). Metastases may be the first clinical evidence of the neoplasm in patients with central nervous system metastatic disease (11), and brainstem involvement may occur (12,13) rarely presenting as INO (14). In a retrospective analysis, brain metastases have been reported to cause INO or the one-and-a-half syndrome in fewer than 1% of cases (2). This may be due to the fact that the posterior circulation accounts for ,25% of total cerebral blood flow. Therefore, hematogenous spread of brain metastasis is less likely to involve the brainstem. Approximately 37% of patients with melanoma eventu-ally develop brain metastasis and, in autopsy reports, 75% of those who died of melanoma developed brain metastasis (15). The imaging features of metastatic melanoma are dis-tinctive because of the presence of melanin and the pro-pensity for hemorrhage. Both hemorrhage and melanin can produce hyperintense appearance on T1-weighted imaging (Fig. 1) while showing signal intensity loss (susceptibility artifact) on susceptibility-weighted imaging (Fig. 2) (16). Our patient illustrates that isolated INO can be the initial presentation of melanoma metastatic to the brain and FIG. 1. A. T1 sagittal magnetic resonance (MRI) imaging shows large solitary mass with peripheral hyperintensity within the pons. After contrast administration, sagittal (B) and axial (C) MRIs demonstrate avid enhancement of the pontine mass. FIG. 2. Susceptibility-weighted axial image reveals dark rim at the margins of the lesion with a nodular appearance on the posterior aspect, signifying presence of blood products. FIG. 3. T1 sagittal magnetic resonance imaging reveals resolution of the mass 4 months after gamma knife radio-surgery. Kumar et al: J Neuro-Ophthalmol 2015; 35: 54-56 55 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. that the extent of metastasis may not be evident from the limited neurological deficits detected clinically. ACKNOWLEDGMENTS The authors are grateful to the late James A. Sharpe, MD, for his guidance and benevolent criticism in preparation of this article. REFERENCES 1. Schraeder PL, Cohen MM, Goldman W. Bilateral internuclear ophthalmoplegia associated with fourth ventricular epidermoid tumor. Case report. J Neurosurg. 1981;54:403-405. 2. Keane JR. Internudear ophthalmoplegia-unusual causes in 114 of 410 patients. Arch Neurol. 2005;62:714-717. 3. Patel AJ, Suki D, Hatiboglu MA, Abouassi H, Shi W, Wildrick DM, Lang FF, Sawaya R. Factors influencing the risk of local recurrence after resection of a single brain metastasis. J Neurosurg. 2010;113:181-189. 4. Bolanos I, Lozano D, Cantu C. Internuclear ophthalmoplegia: causes and long-term follow-up in 65 patients. Acta Neurol Scand. 2004;110:161-165. 5. Arnold AC. Internuclear ophthalmoplegia from intracranial tumor. J Clin Neuroophthalmol. 1990;10:278-286. 6. Troost BT, Martinez J, Abel LA, Heros RC. Upbeat nystagmus and internuclear ophthalmoplegia with brainstem glioma. Arch Neurol. 1980;37:453-456. 7. Leibovitch I, Pakrou D, Selva D, Crompton J. Neuro-ophthalmic manifestations of intracranial cavernous hemangiomas. Eur J Ophthalmol. 2006;16:148-152. 8. Schraeder PL, CM, Goldman W. Bilateral internuclear ophthalmoplegia associated with 4th ventricular epidermoid tumor: case report. J Neurosurg. 1981;54:403-405. 9. 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J Dermatol Sci. 2004;36:71-78. 16. Sloan AE, Nock CJ, Einstein DB. Diagnosis and treatment of melanoma brain metastasis: a literature review. Cancer Control. 2009;16:248-255. STATEMENT OF AUTHORSHIP Category 1: a. Conception and design: S. Kumar; b. Acquisition of data: S. Kumar; c. Analysis and interpretation of data: S. Kumar and M. Sharma. Category 2: a. Drafting the manuscript: S. Kumar and M. Sharma; b. Revising it for intellectual content: S. Shukla and A. N. Sundaram. Category 3: Final approval of the completed manuscript: S. Kumar. 56 Kumar et al: J Neuro-Ophthalmol 2015; 35: 54-56 Clinical Observation Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. |
