Cyclic Esotropia Developing After Strabismus Surgery Richard S. Muchnick, MD; \s=b\
Cyclic
veloped in
Stephanie Sanfilippo, MD; Edward A. Dunlap,
alternate-day esotropia depatient following strabismus intermittent exotropia, which
or a
surgery for resulted in a small overresponse or overcorrection. Surgery, aimed at the deviation on the esotropic days, resulted in binocularity and loss of the cyclic pattern.
(Arch Ophthalmol 94:459-460, 1976)
Cyclicthen,
or
alternate-day strabis¬ first reported in 1958.1 to our knowledge, less
mus was
Since than 50 additional cases have been described in detail.'27 Cyclic strabis¬ mus is a rare condition characterized by a regularly recurring manifest esotropia, usually appearing on alter¬ nate days. On the esotropie days, a constant deviation of 30 to 40 is usual. The condition is unrelated to visual activity, accommodation, fa¬ tigue, or the interruption of fusion, and there is suppression and poor bin¬ ocular function. On the phoric days, only a small latent deviation may be present, and there is physiologic di¬ plopia and full binocular function. The deviation usually appears at 3 to 4 years of age and later becomes a constant esotropia. Treatment is sur¬ gical, aiming at the maximum devia¬ tion. Results are uniformly good, with straight eyes and fusion. The cyclic pattern does not recur and there is no overcorrection on the previously
straight days.
Most of the patients described fol¬ low these general characteristics re¬ markably well. We describe a notable
Submitted for publication Oct 11, 1974. From the Departments of Ophthalmology, New York Hospital-Cornell Medical Center, and the Manhattan Eye, Ear and Throat Hospital (Dr. Muchnick), New York. Reprint requests to 111 E 65th St, New York, NY 10021 (Dr. Muchnick).
exception. To
MD
knowledge, this is the first case report of a patient de¬ veloping cyclic esotropia after previ¬ ous
our
strabismus surgery.
REPORT OF A CASE
creased over the months to ST 20 to 25 and ST' 30 to 35 . No A or V pattern was elicited. There was definite left fixation preference with right amblyopia. Occlusion was only partially successful, with OD vi¬ sual acuity improving to 20/40 + Sensory testing showed right suppression for dis¬ tance and near vision and an absence of .
A 4-year-old right-handed girl, who was the product of a normal 37-week preg¬ nancy, was first seen at the New York Hos¬ pital in November 1960. History disclosed an intermittent exotropia beginning at 1 year of age following a febrile convulsion occurring with measles. There was no fam¬ ily history of strabismus. The findings were X(T) 60 and X(T)' 70 with poor con¬ trol. The near point of convergence was re¬ mote. Ocular rotations were normal. A moderate V pattern was present. Visual acuity could not be tested on the first vis¬ it. However, the patient could alternate fixation. Cycloplegic refraction was OD -15.00=+3.50x90° and OS -13.00 + 3.50x90°. Glasses were prescribed with¬ out a deliberate overcorrection. After wearing the glasses three months, the measurements were X(T) 6 and ' 60 , with an improved near point of conver¬ gence. In March 1961, after wearing glasses nine months, the near deviation ' 35 , with a 30-mm near point of was convergence. The patient reported fusion on the near Worth test. The deviation was better controlled with glasses than without them. In February 1962, the deviation with glasses measured X(T) 60 with poor con¬ trol and X(T)' 50 with good control. The deviation was concomitant. There was an OS fixation preference and acuity with cor¬ rection was OD 20/200 and OS 20/30. At¬ ropine refraction one month later was OD + 3.00 x 90° and OS -11.00 -13.00 + 2.75x90°. In March 1962, the patient underwent a bilateral lateral rectus recession of 8.5 mm. The results of a ten-day postoperative ex¬ amination with glasses showed S 4 and ST' 12 . Subsequent examinations with glasses 3 to 19 months after surgery showed a slightly variable right esotropia for distance and near testing that in=
=
Downloaded From: http://archopht.jamanetwork.com/ by a UQ Library User on 06/20/2015
=
stereopsis. In October 1963, the patient underwent a bilateral medial rectus recession of 3 mm OD and 2 mm OS. Two months postopera¬ tively, the deviation was ST 8 and ST' 15 . The patient was then unavailable for follow-up for more than three years. Between March 1967 and July 1972, the patient was seen 15 times. The measure¬ ments on these visits varied on different days. The distance deviation ranged from ST 10 with RHT 2 to ST 35 with RHT 10 , and the near vision from orthophoria' to ST' 40 with RHT' 12 . An interesting observation is that on seven of the 15 examinations, the deviation for distance and near was 15 or less of esotropia, with an occasional small right hypertropia of 4 or less. On eight of the examinations, the deviation was 18 or greater of esotropia with a variable right hypertropia of 5 or greater. During one examination, the patient asked why her eye would be noticeably deviated on one day and straight on the following day. The patient was examined on four suc¬ cessive days and the measurements with glasses were as follows: first day, ST 10 to 14 with RHT 4 and ST' 4 to 10 (Fig 1); second day, ST 20 to 35 with RHT 6 and ST' 35 with RHT' 10 (Fig 2); third day, ST 10 with RHT 2 to 4 and ortho¬ phoria' to S(T)' 2 ; and fourth day ST 40 with RHT 8 and ST' 40 with RHT' 12 . Personality changes were not noted on any of these days. Both lateral rectus muscles were underactive and the right hyperdeviation seemed to result from underaction of the right superior oblique muscle. The mild amblyopia and strong OS fixation prefer¬ ence persisted. Sensory testing disclosed uncrossed diplopia intermittent with OD suppression when a large deviation was
persisted. There was no cyclic pattern. Re¬ sults of follow-up examinations 12, 18, and 24 months after surgery were essentially the same. There was no progression in the size of the exodeviation and she main¬ tained binocularity.
COMMENT
The most unusual aspect of this is that the cyclic esotropia devel¬ oped following strabismus surgery. Other atypical aspects are the rela¬ tively late age of onset and the high myopia. The satisfactory response to surgery is characteristic. This case il¬ lustrates the need to be aware of this condition and to consider it when varying measurements are obtained. It does not, however, bring us any closer to explaining the cyclic behav¬ ior of the strabismus. Richter8 has reviewed 48-hour cy¬ cles in physiology and behavior that have been found in pediatrie and psy¬ chiatric patients. He cites evidence that the "clock" is located in the hy¬ pothalamus and concludes that 24- to 96-hour cycles, including alternateday esotropia, may all be understood as variations of the fundamental 24hour clock. Roper-Hall and Yapp3 report that a high proportion of their patients showed incomplete or unusual cere¬ bral dominance. They suggest that the alternate-day pattern may be re¬ lated to a hemispheric rivalry and consequent alternation of cerebral dominance. case
Fig 1 .—Cyclic
strabismus.
Straight day.
Fig 2.—Cyclic strabismus. Esotropie day.
References
Fig 3.—Postoperatively. Eyes straight. present. When the deviation was small, the
patient
could intermittently fuse and ap¬ preciate gross stereopsis. However, peri¬ ods of diplopia intermittent with OD sup¬ pression otherwise persisted on the days when the deviation was minimal. On July 25, 1972, the patient underwent a third operation. The left medial rectus muscle was found to insert 7.5 mm from the limbus and was receded to 10 mm from the limbus with a 5-mm conjunctival reces¬ sion over the nasal half of the globe. The
left lateral rectus muscle was found to in¬ sert 15.5 mm from the limbus and was re¬ sected 3 mm and advanced to 7 mm from the limbus. Twelve days postoperatively, 6 and S' 2 . Six the deviation was months after surgery, the deviation was X(T) 4 building up to 16 with RH(T) 6 and X(T)' 4 building up to 25 with RH(T)' 6 (Fig 3). There were periods of binocularity intermittent with crossed di¬ plopia and OD suppression. The OD am¬ blyopia and strong OS fixation preference
Downloaded From: http://archopht.jamanetwork.com/ by a UQ Library User on 06/20/2015
1. Burian HM: Round table discussion, in Allen JH (ed): Strabismus Ophthalmic Symposium II. St. Louis, CV Mosby Co, 1958, p 488. 2. Costenbader FD, Mousel DK: Cyclic esotropia. Arch Ophthalmol 71:180-181, 1964. 3. Roper-Hall MJ, Yapp JMS: Alternate day squint, in The First International Congress of Orthoptics. St. Louis, CV Mosby Co, 1968, pp 262\x=req-\ 271. 4. Chamberlain W: Cyclic esotropia. Am Orthopt J 18:31-34, 1968. 5. Windsor CE, Berg EF: Circadian heterotropia. Am J Ophthalmol 67:565-571, 1969. 6. Costenbader FD, O'Neill J: Cyclic strabismus, in Bellows JG (ed): Contemporary Ophthalmology Honoring Sir Stewart Duke-Elder. Baltimore, Williams & Wilkins Co, 1972, pp 422\x=req-\ 428. 7. Helveston EM: Cyclic Strabismus. Am Orthopt J 22:48-51, 1973. 8. Richter CP: Clock-mechanism esotropia in children. Alternate-day squint. Johns Hopkins Med J 122:218-223, 1968.
Cyclic
veloped in
Stephanie Sanfilippo, MD; Edward A. Dunlap,
alternate-day esotropia depatient following strabismus intermittent exotropia, which
or a
surgery for resulted in a small overresponse or overcorrection. Surgery, aimed at the deviation on the esotropic days, resulted in binocularity and loss of the cyclic pattern.
(Arch Ophthalmol 94:459-460, 1976)
Cyclicthen,
or
alternate-day strabis¬ first reported in 1958.1 to our knowledge, less
mus was
Since than 50 additional cases have been described in detail.'27 Cyclic strabis¬ mus is a rare condition characterized by a regularly recurring manifest esotropia, usually appearing on alter¬ nate days. On the esotropie days, a constant deviation of 30 to 40 is usual. The condition is unrelated to visual activity, accommodation, fa¬ tigue, or the interruption of fusion, and there is suppression and poor bin¬ ocular function. On the phoric days, only a small latent deviation may be present, and there is physiologic di¬ plopia and full binocular function. The deviation usually appears at 3 to 4 years of age and later becomes a constant esotropia. Treatment is sur¬ gical, aiming at the maximum devia¬ tion. Results are uniformly good, with straight eyes and fusion. The cyclic pattern does not recur and there is no overcorrection on the previously
straight days.
Most of the patients described fol¬ low these general characteristics re¬ markably well. We describe a notable
Submitted for publication Oct 11, 1974. From the Departments of Ophthalmology, New York Hospital-Cornell Medical Center, and the Manhattan Eye, Ear and Throat Hospital (Dr. Muchnick), New York. Reprint requests to 111 E 65th St, New York, NY 10021 (Dr. Muchnick).
exception. To
MD
knowledge, this is the first case report of a patient de¬ veloping cyclic esotropia after previ¬ ous
our
strabismus surgery.
REPORT OF A CASE
creased over the months to ST 20 to 25 and ST' 30 to 35 . No A or V pattern was elicited. There was definite left fixation preference with right amblyopia. Occlusion was only partially successful, with OD vi¬ sual acuity improving to 20/40 + Sensory testing showed right suppression for dis¬ tance and near vision and an absence of .
A 4-year-old right-handed girl, who was the product of a normal 37-week preg¬ nancy, was first seen at the New York Hos¬ pital in November 1960. History disclosed an intermittent exotropia beginning at 1 year of age following a febrile convulsion occurring with measles. There was no fam¬ ily history of strabismus. The findings were X(T) 60 and X(T)' 70 with poor con¬ trol. The near point of convergence was re¬ mote. Ocular rotations were normal. A moderate V pattern was present. Visual acuity could not be tested on the first vis¬ it. However, the patient could alternate fixation. Cycloplegic refraction was OD -15.00=+3.50x90° and OS -13.00 + 3.50x90°. Glasses were prescribed with¬ out a deliberate overcorrection. After wearing the glasses three months, the measurements were X(T) 6 and ' 60 , with an improved near point of conver¬ gence. In March 1961, after wearing glasses nine months, the near deviation ' 35 , with a 30-mm near point of was convergence. The patient reported fusion on the near Worth test. The deviation was better controlled with glasses than without them. In February 1962, the deviation with glasses measured X(T) 60 with poor con¬ trol and X(T)' 50 with good control. The deviation was concomitant. There was an OS fixation preference and acuity with cor¬ rection was OD 20/200 and OS 20/30. At¬ ropine refraction one month later was OD + 3.00 x 90° and OS -11.00 -13.00 + 2.75x90°. In March 1962, the patient underwent a bilateral lateral rectus recession of 8.5 mm. The results of a ten-day postoperative ex¬ amination with glasses showed S 4 and ST' 12 . Subsequent examinations with glasses 3 to 19 months after surgery showed a slightly variable right esotropia for distance and near testing that in=
=
Downloaded From: http://archopht.jamanetwork.com/ by a UQ Library User on 06/20/2015
=
stereopsis. In October 1963, the patient underwent a bilateral medial rectus recession of 3 mm OD and 2 mm OS. Two months postopera¬ tively, the deviation was ST 8 and ST' 15 . The patient was then unavailable for follow-up for more than three years. Between March 1967 and July 1972, the patient was seen 15 times. The measure¬ ments on these visits varied on different days. The distance deviation ranged from ST 10 with RHT 2 to ST 35 with RHT 10 , and the near vision from orthophoria' to ST' 40 with RHT' 12 . An interesting observation is that on seven of the 15 examinations, the deviation for distance and near was 15 or less of esotropia, with an occasional small right hypertropia of 4 or less. On eight of the examinations, the deviation was 18 or greater of esotropia with a variable right hypertropia of 5 or greater. During one examination, the patient asked why her eye would be noticeably deviated on one day and straight on the following day. The patient was examined on four suc¬ cessive days and the measurements with glasses were as follows: first day, ST 10 to 14 with RHT 4 and ST' 4 to 10 (Fig 1); second day, ST 20 to 35 with RHT 6 and ST' 35 with RHT' 10 (Fig 2); third day, ST 10 with RHT 2 to 4 and ortho¬ phoria' to S(T)' 2 ; and fourth day ST 40 with RHT 8 and ST' 40 with RHT' 12 . Personality changes were not noted on any of these days. Both lateral rectus muscles were underactive and the right hyperdeviation seemed to result from underaction of the right superior oblique muscle. The mild amblyopia and strong OS fixation prefer¬ ence persisted. Sensory testing disclosed uncrossed diplopia intermittent with OD suppression when a large deviation was
persisted. There was no cyclic pattern. Re¬ sults of follow-up examinations 12, 18, and 24 months after surgery were essentially the same. There was no progression in the size of the exodeviation and she main¬ tained binocularity.
COMMENT
The most unusual aspect of this is that the cyclic esotropia devel¬ oped following strabismus surgery. Other atypical aspects are the rela¬ tively late age of onset and the high myopia. The satisfactory response to surgery is characteristic. This case il¬ lustrates the need to be aware of this condition and to consider it when varying measurements are obtained. It does not, however, bring us any closer to explaining the cyclic behav¬ ior of the strabismus. Richter8 has reviewed 48-hour cy¬ cles in physiology and behavior that have been found in pediatrie and psy¬ chiatric patients. He cites evidence that the "clock" is located in the hy¬ pothalamus and concludes that 24- to 96-hour cycles, including alternateday esotropia, may all be understood as variations of the fundamental 24hour clock. Roper-Hall and Yapp3 report that a high proportion of their patients showed incomplete or unusual cere¬ bral dominance. They suggest that the alternate-day pattern may be re¬ lated to a hemispheric rivalry and consequent alternation of cerebral dominance. case
Fig 1 .—Cyclic
strabismus.
Straight day.
Fig 2.—Cyclic strabismus. Esotropie day.
References
Fig 3.—Postoperatively. Eyes straight. present. When the deviation was small, the
patient
could intermittently fuse and ap¬ preciate gross stereopsis. However, peri¬ ods of diplopia intermittent with OD sup¬ pression otherwise persisted on the days when the deviation was minimal. On July 25, 1972, the patient underwent a third operation. The left medial rectus muscle was found to insert 7.5 mm from the limbus and was receded to 10 mm from the limbus with a 5-mm conjunctival reces¬ sion over the nasal half of the globe. The
left lateral rectus muscle was found to in¬ sert 15.5 mm from the limbus and was re¬ sected 3 mm and advanced to 7 mm from the limbus. Twelve days postoperatively, 6 and S' 2 . Six the deviation was months after surgery, the deviation was X(T) 4 building up to 16 with RH(T) 6 and X(T)' 4 building up to 25 with RH(T)' 6 (Fig 3). There were periods of binocularity intermittent with crossed di¬ plopia and OD suppression. The OD am¬ blyopia and strong OS fixation preference
Downloaded From: http://archopht.jamanetwork.com/ by a UQ Library User on 06/20/2015
1. Burian HM: Round table discussion, in Allen JH (ed): Strabismus Ophthalmic Symposium II. St. Louis, CV Mosby Co, 1958, p 488. 2. Costenbader FD, Mousel DK: Cyclic esotropia. Arch Ophthalmol 71:180-181, 1964. 3. Roper-Hall MJ, Yapp JMS: Alternate day squint, in The First International Congress of Orthoptics. St. Louis, CV Mosby Co, 1968, pp 262\x=req-\ 271. 4. Chamberlain W: Cyclic esotropia. Am Orthopt J 18:31-34, 1968. 5. Windsor CE, Berg EF: Circadian heterotropia. Am J Ophthalmol 67:565-571, 1969. 6. Costenbader FD, O'Neill J: Cyclic strabismus, in Bellows JG (ed): Contemporary Ophthalmology Honoring Sir Stewart Duke-Elder. Baltimore, Williams & Wilkins Co, 1972, pp 422\x=req-\ 428. 7. Helveston EM: Cyclic Strabismus. Am Orthopt J 22:48-51, 1973. 8. Richter CP: Clock-mechanism esotropia in children. Alternate-day squint. Johns Hopkins Med J 122:218-223, 1968.
