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Extraocular muscle cysticercosis presenting as brown syndrome
2. Lim JI, Tessler HH, Goodwin JA. Anterior granulomatous uveitis in patients with multiple sclerosis. Ophthalmology 1991;98:142–145. 3. Kuroiwa Y, Igata A, Itahara K, et al. Nationwide survey of multiple sclerosis in Japan. Clinical analysis of 1084 cases. Neurology 1975;25:845– 851. 4. Moriwaka F, Tashiro K, Fukazawa T, Miyagishi R, Sasamoto Y. A case of multiple sclerosis associated with granulomatous panuveitis. Clin Neurol 1994;34:727–729. 5. Chan ED, Reves R, Belisle JT, Brennan PJ, Hahn WE. Diagnosis of tuberculosis by a visually detectable immunoassay for lipoarabinomannan. Am J Respir Crit Care Med 2000;161: 1713–1719.
vegetables infested with the eggs of the worm, or rarely by autoinfection as a result of reverse peristalsis whereby the eggs retrogradely reach the portal system from the intestines.1 Extraocular muscle cysticercosis is rare.1,2 Isolated reports have demonstrated the different clinical representations of the condition, which include Duane retraction syndrome,1 double depressor palsy,1,2 ophthalmoplegias,2 and Brown syndrome.3 We examined a 20-year-old male patient who presented in October 1999 with complaints of recurrent attacks of conjunctivitis for 6 months before presentation along with double vision in upgaze. There was no history of decrease in vision, trauma, dog bite, or any previous surgery. On general physical, systemic, and neurological examination, no abnormalities were detected. On ocular examination, the uncorrected visual acuity was 20/20 in both eyes. Examination of ocular motility revealed restriction of movement of the right eye in levoelevation. No primary position strabismus was present. No incomitance of ocular movements was found on dextroelevation or in any other gaze. Diplopia charting revealed maximum diplopia in levoelevation. On Hess charting, marked underaction of the right inferior oblique muscle along with overaction of the left superior rectus muscle was found. Forced duction test of the right eye was positive for elevation in adduction (Figure 1). Examination of the rest of the anterior and posterior segment of both eyes was normal. Based on the above findings, a diagnosis of acquired Brown syndrome was made. A contrast-enhanced computed tomographic scan of the head and orbit revealed the presence of a well-defined hyperdense cystic lesion with a scolex within the right superior oblique muscle suggestive of extraocular muscle cysticercosis (Figure 2). The same was confirmed with the help of orbital ultrasonography. However, enzyme-linked immunosorbent assay (ELISA) for cysticercosis was negative. The patient was subsequently started on the benzimidazole carbamate derivative, albendazole (15 mg/kg body weight), along with oral prednisolone (1 mg/kg body weight in tapering doses) for a month. The medication was discontinued after 1 month. A repeat computed tomographic scan performed at 1 month and 2 months after starting treatment revealed the resolution of the cystic lesion. Orbital ultrasonography confirmed the above but revealed enlargement of the right superior oblique muscle, which persisted along with mild restriction of ocular movements in levoelevation. The forced duction test was also mildly positive for restriction of elevation in adduction, which was, however, much less than what it was before starting treatment. The patient did not complain of any conjunctivitis subsequently. There were no complaints of diplopia in primary gaze. However, residual diplopia was present in levoelevation. The patient has currently completed a 6-month follow-up. Acquired motility disorders with different clinical pic-
Extraocular Muscle Cysticercosis Presenting as Brown Syndrome Pramod K. Pandey, MD, Zia Chaudhuri, MS, DNB, MNAMS, and Amit Bhatia, MBBS PURPOSE:
Report of a case of acquired Brown syndrome caused by infestation of the superior oblique muscle by Cysticercus cellulosae. METHOD: Case seen in a referral practice. A 20-year-old man presented with recurrent attacks of conjunctivitis and diplopia in upgaze. Clinical examination of ocular motility established a diagnosis of acquired Brown syndrome of the right eye. Computed tomography of the right orbit unequivocally established the diagnosis of superior oblique muscle cysticercosis. The patient was started on systemic steroids and albendazole in the prescribed doses for a month. RESULT: Serial computed tomography scans of the orbit revealed resolution of the cystic lesion after a month. Clinically, although there was restoration of ocular motility in upgaze, mild restriction of movement of the right eye in levoelevation persisted. However, the patient was symptomatically better with amelioration of the recurrent conjunctivitis and diplopia in primary gaze. CONCLUSION: Extraocular muscle cysticercosis should be considered in the differential diagnosis of acquired motility disorder. The presentation of extraocular muscle cysticercosis as an acquired Brown syndrome is unusual. Response to medical therapy was satisfactory. (Am J Ophthalmol 2001;131:526 –527. © 2001 by Elsevier Science Inc. All rights reserved.)
C
YSTICERCUS CELLULOSAE, THE LARVAL FORM OF THE
tapeworm Taenia solium, occasionally infests humans by way of drinking contaminated water, eating uncooked Accepted for publication Sep 15, 2000. From the Department of Ophthalmology, Guru Nanak Eye Centre, New Delhi, India. Reprint requests to Zia Chaudhuri, MS, DNB, MNAMS, E-310, Purvasha, Anand Lok Society, Mayur Vihar Phase-I, Delhi-110091, India; fax: 91-11-2256931.
526
AMERICAN JOURNAL
OF
OPHTHALMOLOGY
APRIL 2001
FIGURE 1. Clinical photograph of the patient in nine cardinal gazes showing marked restriction of elevation in adduction of the right eye (levoelevation). There is no limitation of movement of either eye in dextroelevation.
may simulate both acquired paralytic or restrictive strabismus,2 although the latter is more common. A computerized literature search revealed this to be the fourth report demonstrating the involvement of the superior oblique muscle2,3,5 by C. cellulosae. An increased awareness of this condition while investigating any cause of acquired motility disorder would probably lead to the condition being diagnosed more often. The management of extraocular muscle cysticercosis has undergone a radical change with the successful results of medical alternatives to surgical excision of the cysts in the form of albendazole and praziquantal,1,2 which have been found to be very effective. However, the long-term changes induced in the muscle as a result of the cysticercus infestation in the form of muscle fibrosis or enlargement need to be studied further. The indications for surgical interventions in the form of muscle surgery for the residual strabismus and the duration after which it can be performed also need to be evaluated. FIGURE 2. Contrast-enhanced computed tomographic scan of the orbit of the same patient showing the presence of a hyperdense cystic lesion with a scolex within (target lesion) the substance of the right superior oblique muscle suggestive of extraocular muscle cysticercosis (shown by an arrow mark). This resulted in the clinical presentation of an acquired Brown syndrome of the right eye, as is shown in Figure 1.
tures because of cysticercosis may be caused by either direct muscle infiltration by the organism or because of cranial nerve palsies as a result of increased intracranial pressure as a sequelae of neurocysticercosis.4 Direct muscle infiltration
VOL. 131, NO. 4
REFERENCES
1. Menon V, Kumar G, Prakash P. Cysticercosis of extraocular muscle. J Pediatr Ophthalmol Strabismus 1994;31:126 –129. 2. Shekhar CG, Lemke BN. Orbital cysticercosis. Ophthalmology 1997;104:1599 –1634. 3. Rao VA, Kawaktra VK, Ratnakar C. Unusual case of acquired inflammatory Brown’s syndrome. Can J Ophthalmol 1987;22: 320 –322. 4. Whiterfield L, Crawston JS, Davey C. Cavernous sinus syndrome associated with neurocysticercosis. Eye 1996;10:642– 643. 5. Brooks AM, Essex WB, West RH. Cysticercus of the superior oblique muscle. Austr J Ophthalmol 1983;11:119 –122.
BRIEF REPORTS
527
vegetables infested with the eggs of the worm, or rarely by autoinfection as a result of reverse peristalsis whereby the eggs retrogradely reach the portal system from the intestines.1 Extraocular muscle cysticercosis is rare.1,2 Isolated reports have demonstrated the different clinical representations of the condition, which include Duane retraction syndrome,1 double depressor palsy,1,2 ophthalmoplegias,2 and Brown syndrome.3 We examined a 20-year-old male patient who presented in October 1999 with complaints of recurrent attacks of conjunctivitis for 6 months before presentation along with double vision in upgaze. There was no history of decrease in vision, trauma, dog bite, or any previous surgery. On general physical, systemic, and neurological examination, no abnormalities were detected. On ocular examination, the uncorrected visual acuity was 20/20 in both eyes. Examination of ocular motility revealed restriction of movement of the right eye in levoelevation. No primary position strabismus was present. No incomitance of ocular movements was found on dextroelevation or in any other gaze. Diplopia charting revealed maximum diplopia in levoelevation. On Hess charting, marked underaction of the right inferior oblique muscle along with overaction of the left superior rectus muscle was found. Forced duction test of the right eye was positive for elevation in adduction (Figure 1). Examination of the rest of the anterior and posterior segment of both eyes was normal. Based on the above findings, a diagnosis of acquired Brown syndrome was made. A contrast-enhanced computed tomographic scan of the head and orbit revealed the presence of a well-defined hyperdense cystic lesion with a scolex within the right superior oblique muscle suggestive of extraocular muscle cysticercosis (Figure 2). The same was confirmed with the help of orbital ultrasonography. However, enzyme-linked immunosorbent assay (ELISA) for cysticercosis was negative. The patient was subsequently started on the benzimidazole carbamate derivative, albendazole (15 mg/kg body weight), along with oral prednisolone (1 mg/kg body weight in tapering doses) for a month. The medication was discontinued after 1 month. A repeat computed tomographic scan performed at 1 month and 2 months after starting treatment revealed the resolution of the cystic lesion. Orbital ultrasonography confirmed the above but revealed enlargement of the right superior oblique muscle, which persisted along with mild restriction of ocular movements in levoelevation. The forced duction test was also mildly positive for restriction of elevation in adduction, which was, however, much less than what it was before starting treatment. The patient did not complain of any conjunctivitis subsequently. There were no complaints of diplopia in primary gaze. However, residual diplopia was present in levoelevation. The patient has currently completed a 6-month follow-up. Acquired motility disorders with different clinical pic-
Extraocular Muscle Cysticercosis Presenting as Brown Syndrome Pramod K. Pandey, MD, Zia Chaudhuri, MS, DNB, MNAMS, and Amit Bhatia, MBBS PURPOSE:
Report of a case of acquired Brown syndrome caused by infestation of the superior oblique muscle by Cysticercus cellulosae. METHOD: Case seen in a referral practice. A 20-year-old man presented with recurrent attacks of conjunctivitis and diplopia in upgaze. Clinical examination of ocular motility established a diagnosis of acquired Brown syndrome of the right eye. Computed tomography of the right orbit unequivocally established the diagnosis of superior oblique muscle cysticercosis. The patient was started on systemic steroids and albendazole in the prescribed doses for a month. RESULT: Serial computed tomography scans of the orbit revealed resolution of the cystic lesion after a month. Clinically, although there was restoration of ocular motility in upgaze, mild restriction of movement of the right eye in levoelevation persisted. However, the patient was symptomatically better with amelioration of the recurrent conjunctivitis and diplopia in primary gaze. CONCLUSION: Extraocular muscle cysticercosis should be considered in the differential diagnosis of acquired motility disorder. The presentation of extraocular muscle cysticercosis as an acquired Brown syndrome is unusual. Response to medical therapy was satisfactory. (Am J Ophthalmol 2001;131:526 –527. © 2001 by Elsevier Science Inc. All rights reserved.)
C
YSTICERCUS CELLULOSAE, THE LARVAL FORM OF THE
tapeworm Taenia solium, occasionally infests humans by way of drinking contaminated water, eating uncooked Accepted for publication Sep 15, 2000. From the Department of Ophthalmology, Guru Nanak Eye Centre, New Delhi, India. Reprint requests to Zia Chaudhuri, MS, DNB, MNAMS, E-310, Purvasha, Anand Lok Society, Mayur Vihar Phase-I, Delhi-110091, India; fax: 91-11-2256931.
526
AMERICAN JOURNAL
OF
OPHTHALMOLOGY
APRIL 2001
FIGURE 1. Clinical photograph of the patient in nine cardinal gazes showing marked restriction of elevation in adduction of the right eye (levoelevation). There is no limitation of movement of either eye in dextroelevation.
may simulate both acquired paralytic or restrictive strabismus,2 although the latter is more common. A computerized literature search revealed this to be the fourth report demonstrating the involvement of the superior oblique muscle2,3,5 by C. cellulosae. An increased awareness of this condition while investigating any cause of acquired motility disorder would probably lead to the condition being diagnosed more often. The management of extraocular muscle cysticercosis has undergone a radical change with the successful results of medical alternatives to surgical excision of the cysts in the form of albendazole and praziquantal,1,2 which have been found to be very effective. However, the long-term changes induced in the muscle as a result of the cysticercus infestation in the form of muscle fibrosis or enlargement need to be studied further. The indications for surgical interventions in the form of muscle surgery for the residual strabismus and the duration after which it can be performed also need to be evaluated. FIGURE 2. Contrast-enhanced computed tomographic scan of the orbit of the same patient showing the presence of a hyperdense cystic lesion with a scolex within (target lesion) the substance of the right superior oblique muscle suggestive of extraocular muscle cysticercosis (shown by an arrow mark). This resulted in the clinical presentation of an acquired Brown syndrome of the right eye, as is shown in Figure 1.
tures because of cysticercosis may be caused by either direct muscle infiltration by the organism or because of cranial nerve palsies as a result of increased intracranial pressure as a sequelae of neurocysticercosis.4 Direct muscle infiltration
VOL. 131, NO. 4
REFERENCES
1. Menon V, Kumar G, Prakash P. Cysticercosis of extraocular muscle. J Pediatr Ophthalmol Strabismus 1994;31:126 –129. 2. Shekhar CG, Lemke BN. Orbital cysticercosis. Ophthalmology 1997;104:1599 –1634. 3. Rao VA, Kawaktra VK, Ratnakar C. Unusual case of acquired inflammatory Brown’s syndrome. Can J Ophthalmol 1987;22: 320 –322. 4. Whiterfield L, Crawston JS, Davey C. Cavernous sinus syndrome associated with neurocysticercosis. Eye 1996;10:642– 643. 5. Brooks AM, Essex WB, West RH. Cysticercus of the superior oblique muscle. Austr J Ophthalmol 1983;11:119 –122.
BRIEF REPORTS
527