CORRESPONDENCE Consecutive cyclic esotropia after surgery for intermittent exotropia Cyclic esotropia is a rare phenomenon in which esotropia and orthophoria alternate over a period of 48 to 96 hours. This phenomenon was first described by Costenbader and Mousel in 1964.1 Most cases of cyclic esotropia have been idiopathic and in children. Adult-onset cyclic esotropia has been reported with optic atrophy, unilateral traumatic aphakia, traumatic sixth nerve palsy, retinal detachment surgery, strabismus surgery, and central nervous system disease.2–10 We report an unusual case of cycling esotropia with onset after a corrective surgery for intermittent exotropia. The cyclic phase persisted for 2 years, after a 48-hour alternate-day pattern. After strabismus surgery for the esotropic angle, the deviation disappeared and the patient remained orthotropic, with 3 months of follow-up to date. A 9-year-old male was referred to our clinic with a complaint of diplopia and inward deviation of right eye on alternate days for 2 years after corrective surgery on his exotropia. The patient had developed an intermittent exotropia at age 5 years. He was treated with correction of the moderate myopia at that time, but the condition progressed and he underwent right lateral rectus recession and medial rectus resection at 7 years of age at local hospital. He was esotropic instantly after surgery and complained of diplopia. He was treated with patching therapy, alternating eyes. A few weeks later, he experienced development of cyclic esotropia, which began to appear on alternate days. On examination, corrected visual acuity was 20/20 in each eye with –4.00 D myopia correction. Sensory testing on orthotropic days demonstrated fusion by Worth 4-Dot test and stereoacuity in the range of 60 arcsec. Ductions and versions were normal, with left eye fixation preference. Motility testing showed an esotropia of 50 PD with glasses in primary gaze. The patient was otherwise healthy and neurologically normal. There was no family history of strabismus, amblyopia, or myopia. The patient was evaluated roughly twice monthly for the next 3 months on consecutive days. On his strabismic days, he had esotropia with 45 to 50 PD of esotropia in primary gaze (Fig. 1A). On “straight” days, the eyes were orthotropic (Fig. 1B). Visual acuity and motility testing were otherwise unremarkable during these visits. A log maintained by the patient’s mother noted an alternating pattern of “good” (orthotropic) and “bad” (esotropic) 24-hour periods, with an equal number of good and bad days in a given month. Three months after the patient’s initial presentation, prolonged alternate patching attempted on a strabismic day did not result in improvement of symptoms. The patient underwent 6-mm left medial rectus recession and 10-mm left lateral rectus tucking. At follow-up 3 months after surgery, the esotropia had disappeared. The patient demonstrated satisfactory alignment and full ocular rotations (Fig. 1C). Sensory
testing demonstrated fusion by Worth 4-Dot test and stereoacuity in the range of 60 arcsec. Childhood-onset cyclic esotropia usually begins around 3 to 4 years of age in the absence of any inciting event. During the strabismus phase, patients have suppression and poor stereopsis. The cycle is broken after strabismus surgery for the esotropic angle. Later-onset cyclic esotropia, which commences after visual maturity, may be triggered by conditions that disrupt fusion or cause diplopia. A 12-year-old female who experienced development of a cyclic esotropia after strabismus surgery for intermittent exotropia at the age of 8 years was reported by Uemura et al.7 Muchnick et al.8 also reported a similar case after overcorrection of intermittent exotropia. The mechanism that underlies the phenomenon is unknown. Because cyclic esotropia often occurs after a fusiondisrupting event,3,6 overcorrection after strabismus surgery may be such an event for our patient. Surgery resulted in orthophoria and loss of the cyclic pattern in this patient like patients reported by Uemura et al.7 and Muchnick et al.8 Good vision in both eyes and normal binocular function might be an important factor underlying good surgical response in these cases. For acquired adult cyclic esotropia with monocular visual impairment, the surgical response varied.10,11 Some patients without binocular vision still showed good response to surgery. This might imply that the good vision and normal binocular function are not essential factors for the surgical response of adult-onset cyclic esotropia. Because we were uncertain whether surgery would be effective in this case, our patient underwent medial rectus recession and lateral rectus tucking rather than conventional
Fig. 1 — A, Fifty PD esotropia on an esotropic day. B, Orthophoria on a nonsquinting day. C, Orthophoria 3 months after surgical correction.
CAN J OPHTHALMOL — VOL. 49, NO. 5, OCTOBER 2014
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Correspondence recession-resection surgery on the horizontal rectus muscles. The cycles disappeared completely after surgery.
Supported by: This work was supported by the Natural Science Foundation of Shandong Province ZR2009CM134.
Luxin Ma, Dong Kong, Zhaoshan Fan, Jie Zhao Provincial Hospital Affiliated to Shandong University, Jinan, P. R. China Correspondence to: Luxin Ma, MD: [email protected] REFERENCES
4. Pillai P, Dhand UK. Cyclic esotropia with central nervous system disease: report of two cases. J Pediatr Ophthalmol Strabismus. 1987;24:237-41. 5. Frenkel RE, Brodsky MC, Spoor TC. Adult-onset cyclic esotropia and optic atrophy. J Clin Neuroophthalmol. 1986;6:27-30. 6. Troost BT, Abel L, Noreika J, Genovese FM. Acquired cyclic esotropia in an adult. Am J Ophthalmol. 1981;91:8-13. 7. Uemura Y, Tomita M, Tanaka Y. Consecutive cyclic esotropia. J Pediatr Ophthalmol. 1977;14:278-80. 8. Muchnick RS, Sanfilippo S, Dunlap EA. Cyclic esotropia developing after strabismus surgery. Arch Ophthalmol. 1976;94:459-60. 9. Riordan-Eva P, Vickers SF, McCarry B, Lee JP. Cyclic strabismus without binocular function. J Pediatr Ophthalmol Strabismus. 1993;30:106-8. 10. Garg SJ, Archer SM. Consecutive cyclic exotropia after surgery for adult-onset cyclic esotropia. J AAPOS. 2007;11:412-3. 11. Di Meo A, Costagliola C, Della Corte M, Romano A, Foria C, Di Costanzo A. Adult-onset cyclic esotropia: a case report. Optom Vis Sci. 2013;90:e95-8.
1. Costenbader FD, Mousel DK. Cyclic Esotropia. Arch Ophthalmol. 1964;71:180-1. 2. Hutcheson KA, Lambert SR. Cyclic esotropia after a traumatic sixth nerve palsy in a child. J AAPOS. 1998;2:376-7. 3. Cole MD, Hay A, Eagling EM. Cyclic esotropia in a patient with unilateral traumatic aphakia: case report. Br. J. Ophthalmol. 1988;72:305-8.
Solitary fibrous tumour of the lacrimal sac presenting with recurrent dacryocystitis Solitary fibrous tumour is a rare spindle cell neoplasm that frequently arises in the pleura and mediastinum.1 This tumour has, however, been reported in numerous other locations and may be misdiagnosed because of variable clinical presentations and histologic appearances. We present a case of solitary fibrous tumour of the lacrimal sac. A 44-year-old female presented with excessive tearing and a slow-growing soft mass in the lacrimal sac fossa, just below the right medial canthal tendon, clinically consistent with a mucocele. She was treated with oral antibiotics in the past for a right dacryocystitis. She presented again in January 2014 with recurrent dacryocystitis. Examination confirmed the presence of a 1.5-cm smooth, firm mass. The left side and the remaining ophthalmic examination were unremarkable. Ultrasound revealed a hypoechoic, solid mass (1.5 1.0 0.9 cm). Increased vascular flow was noted via Doppler ultrasound (Fig. 1). CT scan revealed a discrete lesion in the lacrimal sac fossa consistent with a mucocele of the lacrimal sac. A tumour was not suspected based on clinical or imaging studies. There was no bone erosion. A right dacryocystorhinostomy, insertion of Crawford tube, and partial debulking of middle turbinate were performed. The lacrimal sac wall appeared thickened, consistent with chronic infection. Invasion into surrounding structures was not evident. Histopathologic examination revealed a moderately cellular, spindle cell neoplasm with prominent vascularity. The cells, admixed with thick collagen bands, were arranged in fascicle. The tumour cells exhibited minimal pleomorphism. Necrosis and mitotic figures were not identified (Fig. 1C).
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Can J Ophthalmol 2014;49:e107–e108 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.05.012
Immunohistochemically, the tumour cells were positive for vimentin and CD34, and were negative for S-100, smooth muscle actin, and desmin (Fig. 1D). A reticulin stain confirmed the presence of reticular fibres between tumour cells. The patient had uneventful postoperative recovery and was symptom free at 1 month of follow-up. Approximately 200 cases of lacrimal sac tumours have been reported, the majority being (76%) epithelial tumours.2,3 Malignant epithelial tumours are more common. Squamous papilloma is the most common benign tumour,4 whereas carcinomas (squamous cell, transitional, mucoepidermoid, or adenocarcinoma) are the most common malignant neoplasms.3,4 Nonepithelial tumours of the lacrimal sac, including fibrous histiocytoma,1,5 schwannoma,6 hemangiopericytoma,3 lipoma,1,2 angiosarcoma,7 neurilemmoma,8 and neurofibroma,9 have been reported. Solitary fibrous tumour, melanoma, granulocytic sarcoma, and lymphoma are rare.1,3 Clinically, the tumours often present with epiphora, recurrent dacryocystitis, mass, and sometimes with blood regurgitation.4 These rare neoplasms are prone to misdiagnosis as dacryocystitis, thereby delaying appropriate treatment.6 Solitary fibrous tumour has been described in numerous locations, including the upper respiratory tract, sublingual gland, thyroid gland, breast, periosteum, brain, and the orbit. Although once considered to originate from mesothelial cells, a nonmesothelial origin is now largely accepted.1 By ultrasonography, they often have welldefined borders and a homogeneous echostructure. Histologically, a hemangiopericytoma-like appearance and diffuse sclerosing patterns are the most common patterns.10 They are generally described as proliferation of blandlooking spindled to oval epithelioid cells that form short
CAN J OPHTHALMOL — VOL. 49, NO. 5, OCTOBER 2014
testing demonstrated fusion by Worth 4-Dot test and stereoacuity in the range of 60 arcsec. Childhood-onset cyclic esotropia usually begins around 3 to 4 years of age in the absence of any inciting event. During the strabismus phase, patients have suppression and poor stereopsis. The cycle is broken after strabismus surgery for the esotropic angle. Later-onset cyclic esotropia, which commences after visual maturity, may be triggered by conditions that disrupt fusion or cause diplopia. A 12-year-old female who experienced development of a cyclic esotropia after strabismus surgery for intermittent exotropia at the age of 8 years was reported by Uemura et al.7 Muchnick et al.8 also reported a similar case after overcorrection of intermittent exotropia. The mechanism that underlies the phenomenon is unknown. Because cyclic esotropia often occurs after a fusiondisrupting event,3,6 overcorrection after strabismus surgery may be such an event for our patient. Surgery resulted in orthophoria and loss of the cyclic pattern in this patient like patients reported by Uemura et al.7 and Muchnick et al.8 Good vision in both eyes and normal binocular function might be an important factor underlying good surgical response in these cases. For acquired adult cyclic esotropia with monocular visual impairment, the surgical response varied.10,11 Some patients without binocular vision still showed good response to surgery. This might imply that the good vision and normal binocular function are not essential factors for the surgical response of adult-onset cyclic esotropia. Because we were uncertain whether surgery would be effective in this case, our patient underwent medial rectus recession and lateral rectus tucking rather than conventional
Fig. 1 — A, Fifty PD esotropia on an esotropic day. B, Orthophoria on a nonsquinting day. C, Orthophoria 3 months after surgical correction.
CAN J OPHTHALMOL — VOL. 49, NO. 5, OCTOBER 2014
e107
Correspondence recession-resection surgery on the horizontal rectus muscles. The cycles disappeared completely after surgery.
Supported by: This work was supported by the Natural Science Foundation of Shandong Province ZR2009CM134.
Luxin Ma, Dong Kong, Zhaoshan Fan, Jie Zhao Provincial Hospital Affiliated to Shandong University, Jinan, P. R. China Correspondence to: Luxin Ma, MD: [email protected] REFERENCES
4. Pillai P, Dhand UK. Cyclic esotropia with central nervous system disease: report of two cases. J Pediatr Ophthalmol Strabismus. 1987;24:237-41. 5. Frenkel RE, Brodsky MC, Spoor TC. Adult-onset cyclic esotropia and optic atrophy. J Clin Neuroophthalmol. 1986;6:27-30. 6. Troost BT, Abel L, Noreika J, Genovese FM. Acquired cyclic esotropia in an adult. Am J Ophthalmol. 1981;91:8-13. 7. Uemura Y, Tomita M, Tanaka Y. Consecutive cyclic esotropia. J Pediatr Ophthalmol. 1977;14:278-80. 8. Muchnick RS, Sanfilippo S, Dunlap EA. Cyclic esotropia developing after strabismus surgery. Arch Ophthalmol. 1976;94:459-60. 9. Riordan-Eva P, Vickers SF, McCarry B, Lee JP. Cyclic strabismus without binocular function. J Pediatr Ophthalmol Strabismus. 1993;30:106-8. 10. Garg SJ, Archer SM. Consecutive cyclic exotropia after surgery for adult-onset cyclic esotropia. J AAPOS. 2007;11:412-3. 11. Di Meo A, Costagliola C, Della Corte M, Romano A, Foria C, Di Costanzo A. Adult-onset cyclic esotropia: a case report. Optom Vis Sci. 2013;90:e95-8.
1. Costenbader FD, Mousel DK. Cyclic Esotropia. Arch Ophthalmol. 1964;71:180-1. 2. Hutcheson KA, Lambert SR. Cyclic esotropia after a traumatic sixth nerve palsy in a child. J AAPOS. 1998;2:376-7. 3. Cole MD, Hay A, Eagling EM. Cyclic esotropia in a patient with unilateral traumatic aphakia: case report. Br. J. Ophthalmol. 1988;72:305-8.
Solitary fibrous tumour of the lacrimal sac presenting with recurrent dacryocystitis Solitary fibrous tumour is a rare spindle cell neoplasm that frequently arises in the pleura and mediastinum.1 This tumour has, however, been reported in numerous other locations and may be misdiagnosed because of variable clinical presentations and histologic appearances. We present a case of solitary fibrous tumour of the lacrimal sac. A 44-year-old female presented with excessive tearing and a slow-growing soft mass in the lacrimal sac fossa, just below the right medial canthal tendon, clinically consistent with a mucocele. She was treated with oral antibiotics in the past for a right dacryocystitis. She presented again in January 2014 with recurrent dacryocystitis. Examination confirmed the presence of a 1.5-cm smooth, firm mass. The left side and the remaining ophthalmic examination were unremarkable. Ultrasound revealed a hypoechoic, solid mass (1.5 1.0 0.9 cm). Increased vascular flow was noted via Doppler ultrasound (Fig. 1). CT scan revealed a discrete lesion in the lacrimal sac fossa consistent with a mucocele of the lacrimal sac. A tumour was not suspected based on clinical or imaging studies. There was no bone erosion. A right dacryocystorhinostomy, insertion of Crawford tube, and partial debulking of middle turbinate were performed. The lacrimal sac wall appeared thickened, consistent with chronic infection. Invasion into surrounding structures was not evident. Histopathologic examination revealed a moderately cellular, spindle cell neoplasm with prominent vascularity. The cells, admixed with thick collagen bands, were arranged in fascicle. The tumour cells exhibited minimal pleomorphism. Necrosis and mitotic figures were not identified (Fig. 1C).
e108
Can J Ophthalmol 2014;49:e107–e108 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.05.012
Immunohistochemically, the tumour cells were positive for vimentin and CD34, and were negative for S-100, smooth muscle actin, and desmin (Fig. 1D). A reticulin stain confirmed the presence of reticular fibres between tumour cells. The patient had uneventful postoperative recovery and was symptom free at 1 month of follow-up. Approximately 200 cases of lacrimal sac tumours have been reported, the majority being (76%) epithelial tumours.2,3 Malignant epithelial tumours are more common. Squamous papilloma is the most common benign tumour,4 whereas carcinomas (squamous cell, transitional, mucoepidermoid, or adenocarcinoma) are the most common malignant neoplasms.3,4 Nonepithelial tumours of the lacrimal sac, including fibrous histiocytoma,1,5 schwannoma,6 hemangiopericytoma,3 lipoma,1,2 angiosarcoma,7 neurilemmoma,8 and neurofibroma,9 have been reported. Solitary fibrous tumour, melanoma, granulocytic sarcoma, and lymphoma are rare.1,3 Clinically, the tumours often present with epiphora, recurrent dacryocystitis, mass, and sometimes with blood regurgitation.4 These rare neoplasms are prone to misdiagnosis as dacryocystitis, thereby delaying appropriate treatment.6 Solitary fibrous tumour has been described in numerous locations, including the upper respiratory tract, sublingual gland, thyroid gland, breast, periosteum, brain, and the orbit. Although once considered to originate from mesothelial cells, a nonmesothelial origin is now largely accepted.1 By ultrasonography, they often have welldefined borders and a homogeneous echostructure. Histologically, a hemangiopericytoma-like appearance and diffuse sclerosing patterns are the most common patterns.10 They are generally described as proliferation of blandlooking spindled to oval epithelioid cells that form short
CAN J OPHTHALMOL — VOL. 49, NO. 5, OCTOBER 2014
