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Incomitant Esotropia Following Pterygium Excision Surgery
94
Abstracts
Cohen Syndrome—Report of Seven Cases and Literature Review Elias I. Traboulsi MD, Ray Taban MD, Dina Memoracion-Peralta MD, Andreas Marcotty MD, Adel Mousawi MD, Lihadh I. Al-Gazali MD; Cleveland Clinic Foundation, Cleveland, OH Introduction: Cohen syndrome is a rare, autosomal-recessive syndrome with the following five key features: (1) nonprogressive mental retardation, motor clumsiness, and microcephaly; (2) typical facial features; (3) childhood hypotonia/joint hyperextensibility; (4) retinochoroidal dystrophy and myopia; and (5) isolated neutropenia. The disorder results from mutations in the COH1 gene that plays a role in vesicle-mediated sorting and intracellular protein trafficking. Ophthalmic manifestations include progressive myopia and a progressive chorioretinal degeneration that simulates retinitis pigmentosa. We report seven new patients with Cohen syndrome and review the literature. Methods: Case series and review of the literature. Seven patients from four families underwent complete systemic and ophthalmic evaluations. A meta-analysis of the literature was done and the frequency of the clinical manifestations groups was tabulated. Genetic testing was performed in one patient. Results: All seven patients had typical clinical manifestations of the disease. Six were children and one was an adult, aged 28 years. Progressive myopia was documented in three patients who had multiple examinations. Retinal degeneration was present in all patients. The adult patient had irido-phakodonesis, previously described in a few patients with Cohen syndrome as a result of zonular laxity and spherophakia. One patient had a splicing mutation in the COH1 gene[c.6733-2A⬎G], while the other mutation could not be identified. Conclusions: Cohen is a rare but under-recognized multisystem disorder. Ophthalmologists may be the first to encounter these patients. Ophthalmic manifestations are very significant and one of the main causes of morbidity. Irido-phakodonesis is a late manifestation of the disease and Cohen syndrome should be added to the list of conditions that leads to ectopia lentis. Incomitant Esotropia Following Pterygium Excision Surgery Federico G. Velez MD, Noa Ela-Dalman MD, Arthur L. Rosenbaum MD; Jules Stein Eye Institute, UCLA, Los Angeles, CA Introduction: Strabismus is an uncommon complication following pterygium excision surgery. Direct trauma to rectus muscles and scarring of the conjunctiva-perimuscular connective tissue complex may result in restriction to abduction, incomitant strabismus, and diplopia. Surgical treatment is challenging because it requires a combination of surgery on the conjunctival-perimuscular connective tissue complex and the medial rectus muscle. Purpose: To report the clinical characteristics and management of subjects with incomitant esotropia following unilateral pterigyum excision. Methods: A retrospective review of six consecutive patients who developed incomitant esotropia, limitation to abduction, and diplopia in primary position following unilateral pterigyum excision surgery. Results: The mean preoperative deviation was 6 PD (range 0 to 25 PD) in the primary position and 13.8 PD (range 6 to 25 PD) in the abducting field of the involved eye. Five patients have undergone strabismus surgery; four patients underwent simultaneous surgery on the conjunctiva-perimuscular connective tissue complex and the medial rectus muscle. One subject had conjunctival-perimuscular connective tissue complex surgery alone. Postoperatively all patients resulted in orthotropia in the primary position and the deviation in the abducting field was improved to 5.2 PD (range 0 to 14 PD). Diplopia was eliminated in primary gaze in all patients, but persisted in lateral gaze in two of five patients. Conclusions: Incomitant esotropia is a rare but serious complication following pterigyum excision surgery. Strabismus surgery is effective in improving the primary position deviation though some restriction to lateral gaze may persist. Combination of conjunctiva-perimuscular connective tissue complex and muscle surgery is required to improve ocular alignment and motility.
Journal of AAPOS Volume 10 Number 1 February 2006 Elongation and Selective Weakening of the Superior Oblique Tendon in Brown’s Syndrome Nicholas P. Strong, Gerasimos M. Voros; Royal Victoria Infirmary, Newcastle Upon Tyne, UK Introduction: Several surgical techniques for the management of Brown’s syndrome have been described. However, all these operations affect both the torsional and the depressing effects of the superior oblique. Methods: The management of two patients with Brown’s syndrome is presented. The first had a large vertical deviation without torsion in the primary position. The second had a small-angle vertical deviation but marked torsion in the primary position. Results: In both patients, the superior oblique tendon was separated in the anterior and posterior half. In the first patient, the anterior half was cut nasally, the posterior half was cut temporally, and the two free edges were sutured together. This resulted in 7-mm elongation of the tendon. Postoperatively, the vertical deviation improved and no torsion was induced. In the second patient, the anterior half of the tendon was cut temporally, the posterior half was cut nasally, and the two free edges were sutured together. This resulted in 7-mm elongation of the tendon. Postoperatively the torsion was eliminated, while the vertical deviation was slightly reduced. Discussion: In each case we aimed to elongate the superior oblique tendon, while simultaneously preserving either its torsional or its depressing effect. Therefore, the anterior or the posterior half of the tendon was preserved at its original insertion, respectively, with good postoperative results. Conclusions: In the surgical management of Brown’s syndrome, elongation of the superior oblique tendon with selective preservation of its depressing or torsional effect is possible when indicated. Postoperative Myositis in Reoperated Extraocular Muscles Andrew B. Wolf, Steven M. Archer; University of Michigan, Kellogg Eye Center, Ann Arbor, MI Introduction: The purpose of our study was to report on a series of patients who developed orbital myositis following routine strabismus surgery on previously operated extraocular muscles. Methods: A retrospective medical record review of four patients who developed myositis following routine strabismus surgery on previously operated rectus muscles. All patients had surgery on a previously operated rectus muscle by a single pediatric ophthalmologist between 1998 and 2003. Results: Four patients developed orbital myositis following routine strabismus surgery on previously operated rectus muscles. In the initial patient, the diagnosis of myositis was based on clinical and CT findings. The subsequent three patients had a clinical course similar to the radiologically confirmed case. Symptoms of myositis developed at a mean of 21 days after surgery (range 4 to 34). In all cases, the diagnosis was supported by rapid resolution after treatment with systemic corticosteroids. Conclusion: Orbital myositis can be a rare but dramatic complication of strabismus surgery. Reoperation, as in all cases in this series, may be a predisposing factor.
Abstracts
Cohen Syndrome—Report of Seven Cases and Literature Review Elias I. Traboulsi MD, Ray Taban MD, Dina Memoracion-Peralta MD, Andreas Marcotty MD, Adel Mousawi MD, Lihadh I. Al-Gazali MD; Cleveland Clinic Foundation, Cleveland, OH Introduction: Cohen syndrome is a rare, autosomal-recessive syndrome with the following five key features: (1) nonprogressive mental retardation, motor clumsiness, and microcephaly; (2) typical facial features; (3) childhood hypotonia/joint hyperextensibility; (4) retinochoroidal dystrophy and myopia; and (5) isolated neutropenia. The disorder results from mutations in the COH1 gene that plays a role in vesicle-mediated sorting and intracellular protein trafficking. Ophthalmic manifestations include progressive myopia and a progressive chorioretinal degeneration that simulates retinitis pigmentosa. We report seven new patients with Cohen syndrome and review the literature. Methods: Case series and review of the literature. Seven patients from four families underwent complete systemic and ophthalmic evaluations. A meta-analysis of the literature was done and the frequency of the clinical manifestations groups was tabulated. Genetic testing was performed in one patient. Results: All seven patients had typical clinical manifestations of the disease. Six were children and one was an adult, aged 28 years. Progressive myopia was documented in three patients who had multiple examinations. Retinal degeneration was present in all patients. The adult patient had irido-phakodonesis, previously described in a few patients with Cohen syndrome as a result of zonular laxity and spherophakia. One patient had a splicing mutation in the COH1 gene[c.6733-2A⬎G], while the other mutation could not be identified. Conclusions: Cohen is a rare but under-recognized multisystem disorder. Ophthalmologists may be the first to encounter these patients. Ophthalmic manifestations are very significant and one of the main causes of morbidity. Irido-phakodonesis is a late manifestation of the disease and Cohen syndrome should be added to the list of conditions that leads to ectopia lentis. Incomitant Esotropia Following Pterygium Excision Surgery Federico G. Velez MD, Noa Ela-Dalman MD, Arthur L. Rosenbaum MD; Jules Stein Eye Institute, UCLA, Los Angeles, CA Introduction: Strabismus is an uncommon complication following pterygium excision surgery. Direct trauma to rectus muscles and scarring of the conjunctiva-perimuscular connective tissue complex may result in restriction to abduction, incomitant strabismus, and diplopia. Surgical treatment is challenging because it requires a combination of surgery on the conjunctival-perimuscular connective tissue complex and the medial rectus muscle. Purpose: To report the clinical characteristics and management of subjects with incomitant esotropia following unilateral pterigyum excision. Methods: A retrospective review of six consecutive patients who developed incomitant esotropia, limitation to abduction, and diplopia in primary position following unilateral pterigyum excision surgery. Results: The mean preoperative deviation was 6 PD (range 0 to 25 PD) in the primary position and 13.8 PD (range 6 to 25 PD) in the abducting field of the involved eye. Five patients have undergone strabismus surgery; four patients underwent simultaneous surgery on the conjunctiva-perimuscular connective tissue complex and the medial rectus muscle. One subject had conjunctival-perimuscular connective tissue complex surgery alone. Postoperatively all patients resulted in orthotropia in the primary position and the deviation in the abducting field was improved to 5.2 PD (range 0 to 14 PD). Diplopia was eliminated in primary gaze in all patients, but persisted in lateral gaze in two of five patients. Conclusions: Incomitant esotropia is a rare but serious complication following pterigyum excision surgery. Strabismus surgery is effective in improving the primary position deviation though some restriction to lateral gaze may persist. Combination of conjunctiva-perimuscular connective tissue complex and muscle surgery is required to improve ocular alignment and motility.
Journal of AAPOS Volume 10 Number 1 February 2006 Elongation and Selective Weakening of the Superior Oblique Tendon in Brown’s Syndrome Nicholas P. Strong, Gerasimos M. Voros; Royal Victoria Infirmary, Newcastle Upon Tyne, UK Introduction: Several surgical techniques for the management of Brown’s syndrome have been described. However, all these operations affect both the torsional and the depressing effects of the superior oblique. Methods: The management of two patients with Brown’s syndrome is presented. The first had a large vertical deviation without torsion in the primary position. The second had a small-angle vertical deviation but marked torsion in the primary position. Results: In both patients, the superior oblique tendon was separated in the anterior and posterior half. In the first patient, the anterior half was cut nasally, the posterior half was cut temporally, and the two free edges were sutured together. This resulted in 7-mm elongation of the tendon. Postoperatively, the vertical deviation improved and no torsion was induced. In the second patient, the anterior half of the tendon was cut temporally, the posterior half was cut nasally, and the two free edges were sutured together. This resulted in 7-mm elongation of the tendon. Postoperatively the torsion was eliminated, while the vertical deviation was slightly reduced. Discussion: In each case we aimed to elongate the superior oblique tendon, while simultaneously preserving either its torsional or its depressing effect. Therefore, the anterior or the posterior half of the tendon was preserved at its original insertion, respectively, with good postoperative results. Conclusions: In the surgical management of Brown’s syndrome, elongation of the superior oblique tendon with selective preservation of its depressing or torsional effect is possible when indicated. Postoperative Myositis in Reoperated Extraocular Muscles Andrew B. Wolf, Steven M. Archer; University of Michigan, Kellogg Eye Center, Ann Arbor, MI Introduction: The purpose of our study was to report on a series of patients who developed orbital myositis following routine strabismus surgery on previously operated extraocular muscles. Methods: A retrospective medical record review of four patients who developed myositis following routine strabismus surgery on previously operated rectus muscles. All patients had surgery on a previously operated rectus muscle by a single pediatric ophthalmologist between 1998 and 2003. Results: Four patients developed orbital myositis following routine strabismus surgery on previously operated rectus muscles. In the initial patient, the diagnosis of myositis was based on clinical and CT findings. The subsequent three patients had a clinical course similar to the radiologically confirmed case. Symptoms of myositis developed at a mean of 21 days after surgery (range 4 to 34). In all cases, the diagnosis was supported by rapid resolution after treatment with systemic corticosteroids. Conclusion: Orbital myositis can be a rare but dramatic complication of strabismus surgery. Reoperation, as in all cases in this series, may be a predisposing factor.