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Rare cases offer new possibility in etiology of pediatric secondary cyclic esotropia
A pediatric case revealed an abnormality that is the first of its kind reported, a clinician said.
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Secondary cyclic esotropia in pediatric patients is a rare condition that may be related to an abnormality at the insertion site of the lateral rectus muscle tendon, a physician found.
“Each [of the] three cases [we treated] have different etiology, and the different course of disease and the reason for the periodicity of the symptom is not clear,” Mayu Sawada, MD, said at the Japan Clinical Ophthalmology annual meeting.
“There is a possibility that abnormality at [the] tendon insertion site is involved in the cyclic esotropia after the strabismus procedure. We need to examine for the abnormality at the tendon insertion site, especially when the strabismus was overcorrected,” she said.
Dr. Sawadaand colleagues treated three cases of pediatric secondary cyclic esotropia with strabismus surgery at the maximum strabismus angle. Good long-term results were reported in two of the three patients. The third patient exhibited an abnormality at the lateral rectus muscle tendon’s insertion site after exotropia surgery and is continuing treatment after esotropia recurred. This case was one of the first of its kind reported.
Cyclic esotropia is characterized by a periodic change in the affected eye from strabismus to orthophoria over a period of 48 to 96 hours. Its reported incidence is one in 3,000 to 4,000 strabismus cases. Most often, patients have good binocular vision, but there is a transition to constant esotropia within months to several years after surgery. Generally, patients are operated on at the maximum strabismus angle when the condition becomes evident.
The condition’s pathogenesis is not yet known. Possible causes reported in the literature include ocular surgery, blocking by eye patch, high fever, injuries including head injury, abducens nerve paralysis, transition from accommodative esotropia, injury after abducens nerve paralysis, and tumor.
First two cases
Dr. Sawada presented two cases in which cyclic esotropia occurred after primary disease and exotropia surgery. The first case involved a boy with a history of idiopathic abducens nerve paralysis of the left eye at 1 year of age. The child later developed cyclic esotropia.
The left eye gradually showed periodicity over the course of 1 month from first demonstrating esotropia. At 2 years, the periodicity was replaced by constant esotropia. The child was operated on at 29 months old by the Krimsky method for esotropia of 30 D and for the overreaction of the right inferior oblique. Bilateral medial rectus muscle recession and resection of the right inferior oblique were performed. However, strabismus recurred 5 months postoperatively, and the visual acuity of the right eye was poor.
At 10 months postop, cycloplegic refraction showed hyperopia and astigmatism, so the patient received prescription eyeglasses and eye patch therapy. As a result, the eye position became orthophoric, and visual acuity showed improving tendency.
The second case involved a girl who developed cyclic esotropia after exotropia surgery. She was diagnosed with exotropia at about the age of 18 months and was operated on for 35 D exotropia at 6 years old. Cyclic esotropia was observed after surgery, gradually becoming constant. After 3 years, at age 9, the patient underwent a bilateral medial rectus muscle recession procedure for esotropia with distance deviation of 45 D and near deviation of 50 D.
At 5 years postop, the patient retained good eye position and stereoscopic sight.
Third case
Dr. Sawada also presented the case of a boy with cyclic esotropia after exotropia surgery and an abnormality at the tendon insertion site resulting from the exotropia operation. This was the first ever report of abnormal tendon insertion site.
The patient was diagnosed with exotropia at age 3 years and was operated on for 25 D of exotropia with a bilateral lateral rectus muscle recession procedure.
At 3 months postop, the left eye demonstrated 30 D esotropia, and 5 months after the first surgery, the patient underwent a lateral rectus muscle advancement procedure on the left eye. However, esotropia remained after surgery, with distance deviation of 4 D and near deviation of 10 D with the limited extortion of right eye and bilateral overreaction of the inferior oblique muscle. This case exhibited fluctuation in the strabismus angle.
The patient underwent a bilateral lateral rectus muscle advancement procedure for esotropia at the age of 5 years and 7 months. Examination of the surgical wound from the previous procedure showed that the tissue at the insertion site of the recessed lateral rectus muscle in both eyes was stretched posteriorly, and the muscle and sclera were joined by tendon-like tissue.
As a result of surgery, the eye position became orthophoric at 1 day postop, and the eye position had been maintained favorably at 5 months postop. Stereoscopic vision was also improving. - by Aya Tokaji
Reference:
Hutcheson KA, Lambert SR. Cyclic esotropia after a traumatic sixth nerve palsy in a child. J AAPOS.1998;2(6):376-377.
For more information:
Mayu Sawada, MD, can be reached at 1-20-1, Handayama, Higashi-ku, Hamamatsu, Shizuoka, 431-3192 Japan; +81-53-435-2111; email: m.sawada@hama-med.ac.jp.
Disclosure: No products or companies are mentioned that would require financial disclosure.