Spontaneous resolution of Infantile Esotropia

Spontaneous resolution of Infantile Esotropia

Spontaneous Resolution of Infantile Esotropia Mi Ah Shon, MD,a Kyoung Hoon Hahm, MD,a Seung Han Han, MD,b and Jeong-Min Hwang, MDc Purpose: To report ...

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Spontaneous Resolution of Infantile Esotropia Mi Ah Shon, MD,a Kyoung Hoon Hahm, MD,a Seung Han Han, MD,b and Jeong-Min Hwang, MDc Purpose: To report the spontaneous resolution of infantile esotropia in 3 patients. Methods: The clinical histories and the results of ophthalmologic examinations in 3 patients with infantile esotropia were reviewed and analyzed with reference to the literature. Results: All 3 patients with infantile esotropia were diagnosed with 25 to 30 PD of esotropia before the age of 6 months. All of them had insignificant refractive errors. Against medical advice, they were not brought in for follow-up examinations. At the age of 34 months to 59 months, the esotropia of the patients had changed into exophoria, esophoria less than 4 PD, or orthophoria. All patients eventually showed dissociated vertical deviation and overaction of the inferior oblique muscles. Of the 2 cooperative patients, 1 consistently identified Titmus stereograms with 3000 seconds of arc and fused Worth 4 dots at near and at distance. The other patient could not identify stereo targets and suppressed one eye on Worth 4 testing at distance, though she showed no suppression at near. Conclusion: In these cases, infantile esotropia with a relatively small angle may spontaneously resolve without any treatment. However, all these patients showed poor stereoacuity, dissociated vertical deviation, and overaction of the inferior oblique muscles. (J AAPOS 2001;5:44-7)

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nfantile esotropia is an esodeviation with onset before the age of 6 months and is usually characterized by a large angle and cross fixation.1 More broadly inclusive accounts of infantile esotropia have suggested a lack of uniformity in patients with the condition,2-4 as well as a variability in their course after undergoing surgical alignment.2-6 Costenbader2 reported that small deviations less than 24 PD and moderate deviations between 25 to 29 PD were found in 21.2% and in 26.4% of infantile esotropic patients, respectively. Thus, it is apparent that the range of initial deviation is considerable, though the large deviations predominate. It may not be clear during early examination which patients will have spontaneous improvement in their misalignment. Much of the recent discussion of infantile esotropia has focused on the optimal time for surgical correction.2-4 A better understanding of the variations in its course should improve our current treatment protocols.

Even though we cannot be certain that these small groups of patients would have developed exotropia (overcorrection) if they had early surgery, some chance of overcorrection might exist. As far as we know, spontaneous resolution of infantile esotropia has been the subject of brief comment6 but has never been reported with photographic documentation. We present 3 patients in which the angle of infantile esotropia decreased without any treatment.

PATIENTS AND METHODS We included cases of spontaneous resolution of infantile esotropia during a 5-year period (1995-1999) according to the following criteria: onset of constant esotropia before the age of 6 months; no prior surgery; no prior orthoptic treatment; no significant accommodative component; no evidence of neurologic dysfunction; no evidence of ocular pathology other than strabismus; and a follow-up period of at least 1 year.

REPORT OF CASES From the Department of Ophthalmology, Chunchon Sacred Heart Hospital, Hallym University, Chunchon, Koreaa; the Department of Ophthalmology, Yongdong Severance Hospital, Yonsei University, Seoul, Koreab; and Department of Ophthalmology, Seoul Municipal Boramae Hospital, Seoul National University, Seoul, Korea.c Submitted April 15, 2000. Revision accepted August 18, 2000. Reprint requests: Jeong-Min Hwang, MD, Department of Ophthalmology, Seoul Municipal Boramae Hospital affiliated with Seoul National University Hospital, 395, Sindaebang-2-dong, Dongjak-ku, Seoul 156-012, Korea (e-mail: [email protected]). Copyright © 2001 by the American Association for Pediatric Ophthalmology and Strabismus. 1091-8531/2001/$35.00 + 0 75/1/111013 doi:10.1067/mpa.2001.111013

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Case 1 A 4-month-old girl presented to 2 of the authors (M.A.S., K.H.H.) with esotropia. She fixed and followed with either eye. At near, she had 30 PD of esotropia, as measured with the Krimsky method (Figure 1). Ductions and versions were full. Cycloplegic refraction showed a mild hypermetropia of +1.00 D sphere OU. The anterior segment and fundi were normal (Table). Subsequently, the patient did not return for follow-up examination until the age of 59 months, when she had 4 Journal of AAPOS

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FIG 1. Photograph of case 1 at the age of 4 months shows right esotropia.

FIG 3. Photograph of case 2 at the age of 4 months shows left esotropia.

A

A B

B C FIG 2. Ocular versions of case 1 at the age of 59 months show 4 PD of exophoria in the primary position and bilateral inferior oblique muscle overaction.

PD of exophoria (Figure 2, B) and 10 PD of dissociated vertical deviation (DVD) OS in the primary and upgaze positions, and twelve PD of esotropia in the downgaze position at distance, as measured with the alternate prism cover test. Her best corrected visual acuity was 20/25 OD and 20/40 OS. Ductions were full and versions showed overaction of both inferior oblique muscles (Figure 2, A and C). She fused Worth 4 dots at near. She consistently identified Titmus stereograms (Titmus Optical Company, Petersburg, Va, USA) with 3000 seconds of arc. At the age of 63 months, her left visual acuity improved to 20/25 with patching of the right eye for a month, and she underwent bilateral inferior oblique muscle myectomies.

C FIG 4. Ocular versions of case 2 at the age of 40 months show orthophoria in primary position and bilateral inferior oblique muscle overaction.

At the age of 6 years, she had 5 PD of esophoria, 2 PD of DVD OD, and 6 PD of DVD OS in the primary position, as measured with the alternate prism cover test. Her best corrected visual acuity was 20/20 OD and 20/25 OS. She had a mild myopia of 1.00 D sphere with cycloplegic refraction. She fused both the Worth 4 dots at near and at distance and the streaks with Bagolini striated glass test. She consistently identified Titmus stereograms with 3000 seconds of arc.

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B

C

FIG 5. Ocular versions of case 3 at the age of 4 years show orthophoria in the primary position and a mild overaction of both inferior oblique muscles, more prominent in the left eye.

Case 2 A 4-month-old boy presented to 2 of the authors (M.A.S., K.H.H.) with esotropia. He fixed and followed with either eye. At near, he had 30 PD of left esotropia, as measured with the Krimsky method (Figure 3). Ductions and versions were full. Cycloplegic refraction showed a mild myopic astigmatism of plano –1.00 × 90 OU. The anterior segment and fundi were normal (Table). The patient did not return for follow-up until the age of 34 months. At that time, he had 4 PD of esophoria, 18 PD of DVD OD, and 10 PD of DVD OS in the primary position at distance, as measured with the alternate prism cover test. Ductions were full and versions showed overaction of both inferior oblique muscles. At the age of 40 months, he was orthophoric with both eyes open (Figure 4, B) and had 14 PD of DVD OD and 7 PD of DVD OS in the primary position, as measured with the alternate prism cover test. Ductions were full and versions showed overaction of both inferior oblique muscles (Figure 4, A and C). He had a mild myopia of –0.50 D sphere OD and –0.75 D sphere OS with cycloplegic refraction. He was not cooperative enough to perform Worth 4 dot test or Titmus test. The 4 PD base-out test revealed a suppression scotoma OD. Case 3 A 3-year-old girl presented to 1 of the authors (J.M.H.) with a history of esotropia. She was first noted to constantly cross her eyes at the age of 3 months. At the age of 5 months, she was diagnosed by another author (S.H.H.) to have 25 PD of right esotropia by the Krimsky method with a low hyperopic refractive error of +0.50 D sphere. At that time, she fixed and followed with either eye. Ductions and versions were full. On 3 more examinations in her first year, she consistently showed 25 to 30 PD of esotropia with the Krimsky method. Against medical recommendation, she did not undergo any surgery (Table). At the age of 39 months, she was found to be orthophoric at distance and at near, as measured with the alternate prism cover test by 1 of the authors (J.M.H.). Her best corrected visual acuity was 20/30 OU. Ductions were full and versions showed a mild overaction of both inferior oblique muscles, more prominent in the left eye. Cycloplegic

refraction indicated the presence of a mild hypermetropia of +0.25 –0.50 × 180 OD and +0.25 D sphere OS. The anterior segment and fundi were normal. The patient could not identify flies or animals in random dot stereograms or Titmus stereograms. She fused Worth 4 dots at near and at distance. Monocular optokinetic nystagmus was asymmetric in each eye. At the age of 4 years, she was still orthophoric at distance and at near, as measured with the alternate prism cover test (Figure 5). All the ophthalmologic findings remained the same. At the age of 5 years, the visual acuity was 20/25 OU. At distance, she had 4 PD of exophoria and 6 PD of DVD OS and 2 PD of exophoria at near, as measured with the alternate prism cover test. Ductions were full and versions showed a mild overaction of both inferior oblique muscles, more prominent in the left eye. The patient could not identify flies or animals in random dot stereograms or Titmus stereograms. She fused Worth 4 dots at near and suppressed the right eye at distance.

DISCUSSION There have been no data regarding the prevalence of spontaneous resolution of esotropia. Spontaneous divergence to an acceptable alignment has seldom been mentioned.2,6-9 Robb and Rodier6 briefly described the case of a boy with 25 PD of infantile esotropia who straightened at the age of 6 years with some binocular vision and gross stereopsis. Then by the age of 9 years, he had developed an intermittent exotropia of 12 PD.6 Detailed history and pictures were not presented. Clarke and Noel7 reported on 3 cases with 30 to 40 PD of infantile esotropia that spontaneously developed into esotropia of approximately 10 PD without surgery or the use of glasses.7 Forrest8 also reported on a patient with 25 to 30 PD of infantile esotropia that spontaneously developed into esophoria of 15 PD with orthoptic treatment. None of them became as straightened as our patients. All the patients in Clarke and Noel’s series developed DVD and 2 of them developed inferior oblique overaction. Birch et al9 reported that a few infants who initially had small angle or variable angle esotropia showed resolution to orthophoria. They concluded that infants with constant esotropia of 40 PD or greater are valid candidates for surgical treatment.9 Good et al10 described 20 adults with early onset of esotropia who did not receive any treatment. They noted

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TABLE. Clinical data in 3 cases with resolution of infantile esotropia Case No. 1 Esotropia Age of onset of esotropia (mo) Age at first diagnosis (mo) Ocular alignment Refractive error at first visit Orthophorization Age at diagnosis of orthophoria (mo) Ocular alignment Final visit Age at final visit (mo) Ocular alignment Visual acuity Refractive error DVD Age at first diagnosis (mo) Amount Inferior oblique overaction Age at first diagnosis (mo) Amount Amblyopia Stereoacuity (seconds of arc) Worth 4 dot

2

3

Birth 44 Esotropia 30 PD +1.00 D sphere OU

2 5 Esotropia 30 PD Plano –1.00 × 90 OU

59 Exophoria 4 PD DVD 10 PD OS

34 Esophoria 4 PD DVD (18 PD OD, 10 PD OS)

39 Orthophoria

67 S/P BIO myectomy (63 mo); esophoria 5 PD; DVD 2 PD OD, 6 PD OS 20/20 OU –1.00 D sphere OU

40 DVD 14 PD OD, 6 PD OS

66 Exophoria 4 PD; DVD 6 PD OS 20/25 OU +0.25 –0.50 × 180 OD; +0.25 sphere OS

59 10 PD OS

34 18 PD OD, 10 PD OS

66 6 PD OS

59 +3 OD, +4 OS 59 mo: Mild (20/25 OD, 20/40 OS); 67 mo:– 3000 N, fusion; Dis, fusion

34 +3 OD, +2 OS – Not cooperative Not cooperative

39 +1 OU – nil 39-49 mo: N, fusion; Dis, fusion; 66 mo: N, fusion; Dis, suppress OD

Fix & follow Plano –1.00 × 90 OU

3 Esotropia 25-30 PD +0.5 D sphere OU

N, near; Dis, distance.

that the incidences of amblyopia, DVD, and inferior oblique overaction were very low (15%, 10%, and 5%, respectively) compared with the control group who underwent the surgery. They also suggested that DVD or inferior oblique overaction is uncommon in untreated infantile esotropes and may be expressed after surgical alignment. However, all or most of our patients and Clarke and Noel’s patients also developed DVD and inferior oblique overaction.8 Development of DVD and inferior oblique overaction may be related to reduction of the amount of esotropia, not to surgery itself. In addition to infantile esotropia, several other forms of esotropia can be found during infancy. These include bilateral sixth nerve palsy and refractive accommodative esotropia. Our patients showed normal ductions and versions and had a low hyperopia or myopic astigmatism, thereby ruling out bilateral sixth nerve palsy or early onset refractive accommodative esotropia. In conclusion, those rare cases of infantile esotropia that have a relatively small angle may spontaneously resolve without any treatment. The cases we report showed poor stereoacuity, DVD, and overaction of the inferior oblique muscles. These patients offered us the unusual opportunity to evaluate the natural history of infantile esotropia and to assess binocular outcome. We do not recommend withholding treatment, but it is necessary

to note that the esotropia may decrease or even resolve spontaneously. When performing very early surgery for infantile esotropia, the rare possibility of spontaneous resolution should be considered. Further studies must be made in a large number of infantile esotropia patients. References 1. Nelson LB, Wagner RS, Simon JW, Harley RD. Congenital esotropia. Surv Ophthalmol 1987;31:363-83. 2. Costenbader FD. Infantile esotropia. Trans Am Ophthalmol Soc 1961;59:397-429. 3. Ing M, Costenbader FD, Parks MM, Albert DG. Early surgery for congenital esotropia. Am J Ophthalmol 1966;61:1419-27. 4. Foster RS, Paul OT, Jampolsky A. Management of infantile esotropia. Am J Ophthalmol 1976;82:291-9. 5. Wright KW, Edelman PM, McVey JH, Terry AP, Lin M. Highgrade stereo acuity after early surgery for congenital esotropia. Arch Ophthalmol 1994;112:913-9. 6. Robb RM, Rodier DW. The variable clinical characteristics and course of early infantile esotropia. J Pediatr Ophthalmol Strabismus 1987;24:276-81. 7. Clarke WN, Noel LP. Vanishing infantile esotropia. Can J Ophthalmol 1982;17:100-2. 8. Forrest EB. Treating infant esotropia: a case report. Am J Optom Physiol Opt 1978;55:463-5. 9. Birch E, Stager D, Wright K, Beck R. The natural history of infantile esotropia during the first six months of life. J AAPOS 1998; 2:325-9. 10. Good WV, da Sa LC, Lyons CJ, Hoyt CS. Monocular visual outcome in untreated early onset esotropia. Br J Ophthalmol 1993;77:492-4.