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   Table of Contents      
ORIGINAL ARTICLE
Year : 2007  |  Volume : 55  |  Issue : 2  |  Page : 117-120

Clinical characteristics of spontaneous late-onset comitant acute nonaccommodative esotropia in children


Department of Pediatric Ophthalmology and Strabismus, Bombay City Eye Institute and Research Centre, 5 Victor Villa, Babulnath Road, Mumbai - 400 007, India

Date of Submission12-Dec-2005
Date of Acceptance26-Jul-2006

Correspondence Address:
Mihir Kothari
Jyotirmay Eye Clinic for Children and Squint and Pediatric Low Vision Center, 205, Ganatra Estate, Pokhran Road No 1, Khopat, Thane West - 400 601, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0301-4738.30705

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  Abstract 

Purpose: To describe the clinical characteristics of spontaneous, late-onset comitant acute, nonaccommodative esotropia (ANAET) in children.
Materials and Methods: Clinical characteristics of a cohort of patients under 16 years of age with ANAET were studied retrospectively.
Results: Of 15 patients eight were females. Mean age was 7.15 years (range 2.5-13, SD 3.34). Mean age of the onset of deviation was 3.2 years (range 1.5-9, SD 2.26). Mean duration of strabismus was 36 months (range 3-132, SD 43). History of a precipitating event was present in five patients (33.3%). Mean cycloplegic refraction was 1.84 diopter sphere (range -5.75 to +7.25, SD 3.55). Mean esodeviation for near and distance fixation was 40 prism diopter (range 15-90, SD 23.9). None had near/distance disparity of more than 5 prism diopter. Amblyopia was present in 13 cases (87%). Strabismus surgery was performed for eight patients. Five patients had orthophoria and three were aligned within 8 prisms esodeviation.
Conclusion: ANAET is more common than previously reported, has a variable time of onset and high incidence of amblyopia. Timely management would avert emergence of amblyopia and vertical incomitance thereby promising better binocular outcome.

Keywords: Acute-onset, amblyopia, esotropia, late-onset, spontaneous, strabismus


How to cite this article:
Kothari M. Clinical characteristics of spontaneous late-onset comitant acute nonaccommodative esotropia in children. Indian J Ophthalmol 2007;55:117-20

How to cite this URL:
Kothari M. Clinical characteristics of spontaneous late-onset comitant acute nonaccommodative esotropia in children. Indian J Ophthalmol [serial online] 2007 [cited 2019 Aug 20];55:117-20. Available from: http://www.ijo.in/text.asp?2007/55/2/117/30705

Clinical characteristics of patients with acute nonaccommodative esotropia

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Clinical characteristics of patients with acute nonaccommodative esotropia

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Prevalence of strabismus ranges from 3-5%,[1],[2] esodeviations being more common than exodeviations (prevalence ratio 5:2).[3],[4] However, geographical and ethnic variations in this pattern are known.[4],[5],[6] Amongst esodeviations, accommodative esotropia is the commonest type followed by congenital or infantile esotropia.[7] The prevalence of spontaneous late-onset comitant acute nonaccommodative esotropia (ANAET) (after one year) remains unknown but is certainly considered rare.[8] Previous investigators[8],[9],[10],[11],[12],[13],[14],[15] have classified ANAET into five subtypes-Type 1 / Swan type: Esotropia due to the disruption of the fusion (precipitated by monocular occlusion), Type 2 / Burian-Francesschetti type characterized by minimal hypermetropia and diplopia that is often associated with physical or psychological stress, Type 3 / Bielchowsky type: occurs in adults with myopia, Type 4 / Refractive-accommodative type characterized by high hypermetropia that can be adequately controled with the refractive correction alone, Type 5: Associated with intracranial pathology, most commonly a posterior fossa lesion.

We herein report the clinical characteristics of 15 children with ANAET presented within a period of 17 months with the history of sudden onset of deviation after age of one year not associated with occlusion, intracranial pathology or accommodative component. To our knowledge this is the first report on ANAET in children from India and we believe this variety of strabismus occurs more frequently than reported in the medical literature.


  Materials and Methods Top


This retrospective cohort study included 15 consecutive patients presenting in the department of Pediatric Ophthalmology and Strabismus of a tertiary eye centre in Western India between April 2003 and August 2004. The medical records of the patients diagnosed to have ANAET who fulfilled the following criteria were included in the study.

  1. Age under 16 years,
  2. Sudden onset of strabismus (within a few hours to days and photographic evidence of absence of strabismus before the history of onset of esotropia),
  3. Convergent and comitant strabismus,
  4. Age of onset after one year,
  5. No accommodative component.


Following data were abstracted from the medical records:

  • Age of the patient,
  • Age at the onset of deviation,
  • Duration of deviation,
  • Gender,
  • Prior debilitating illness,
  • Association with vaccination or teething,
  • Vision,
  • Angle of deviation for near and distance fixation,
  • Pattern of deviation,
  • Cycloplegic refraction and
  • Surgical outcome.


A strabismologist examined all the patients. A detailed systemic history and clinical examination was done to rule out neurological lesion.

The data were entered in a Microsoft excel data sheet. Standard deviation and 95% confidence intervals were calculated.


  Results Top


Of 15 patients [Table - 1] eight were females. Mean age was 7.15 years (range 2.5-13, SD 3.34). Mean age of the onset of deviation was 3.2 years (range 1.5-9, SD 2.26). Mean duration of strabismus was 36 months (range 3-132, SD 43). History of precipitating event was present in five cases (33.3%). Each of these had febrile illness. One had associated teething and another one had associated febrile seizure with normal CT scan of the brain. Mean cycloplegic refraction was 1.84 diopter sphere (DS) (range -5.75 to +7.25, SD 3.55). Anisometropia of 1.25 DS was present in one. Mean esodeviation for near and distance fixation was 40 prism diopter (PD) (range 15-90, SD 23.9). The majority (80%, 12 patients) had a deviation measuring over 30 PD. None had near/distance disparity of more than 5 PD. Vertical incomitance was present in four cases that was equal to 4 PD. Significant pattern with inferior oblique over action (30 PD of V pattern in patient number 12) was present in one. Amblyopia was present in 13 cases (87%); unilateral (strabismic amblyopia) in eight, with the left eye being more involved (six patients), mixed (anisometropic with strabismic) amblyopia in one and strabismic amblyopia with ametropic amblyopia in four. The density of the amblyopia was mild (vision in amblyopic eye better than 20/60) in all cases. Strabismus surgery was advised for all the patients, eight patients underwent the surgery. One patient with large 30 PD V pattern also underwent simultaneous bilateral 10 mm recession of the inferior oblique muscles (Patient Number 12). Postoperatively five patients had orthophoria and two were aligned within less than 8 PD of esotropia and one had intermittent esotropia less than 8 PD at the last follow-up (mean 8.7 months, range 4-108 weeks). Four patients who underwent strabismus surgery have completed more than 10 months follow-up without developing any neurological lesion. Patient number 12 had presence of binocular single vision for near and distance on Worth 4 dot test, patient Number 3 had fusion response for the near only and patient Number 1 had constant suppression in the squinting eye. Sister of patient Number 1 developed esotropia at four months of age. The parents of both these children were orthophoric and the perinatal history was not significant. She underwent bilateral medial rectus recession after three months of occlusion therapy at the age of 14 months achieving orthophoria.


  Discussion Top


The ANAET is considered less common than accommodative and congenital esotropia.[7],[8] However, we examined 15 patients within a span of 17 months and we have continued to see more number of patients raising the possibility of a higher incidence of this strabismus in this part of the world.

None of the patients in our series had history of monocular occlusion preceding the onset of strabismus. They neither had any intracranial abnormality on presentation nor developed any later. None were myopic adults to be labeled as Bielchowsky's type of ANAET. Our inclusion criteria effectively excluded accommodative esotropia. Hence our patients seem to fall in the subgroup that resembles Type 2 (Burian-Francesschetti type) ANAET. In Burian's paper[10] the age group was 11-72 years, patients had low hypermetropic errors, they complained of diplopia, angle of deviation ranged between 20 to 60 PD and had excellent functional outcome after the strabismus surgery. Our study had patients in a younger age group and none of the parents could recall their child complaining of diplopia. This could be a recall bias in some, rapidity of suppression in very young children or inability on the part of very young children to complain diplopia. This could also happen if these patients had decompensation of a preexisting microtropia.[16] Only the absence of bifoveal fusion before the onset of esoptropia could have confirmed this notion. A photographic evidence of absence of strabismus could be deceptive in those cases. However, in a previous report by Spierer[17] where ten adults with ANAET were surgically aligned, none had decompensated microtropia as all gained normal stereopsis. The duration between the onset of strabismus and realignment may be important.

The incidence of unilateral amblyopia was found to be high in this study. Once amblyopia is established in the patients with ANAET there is always a suppression scotoma. However, this is expected to have taken some time after the onset of esotropia, unless the patients with the ANAET had a preexisting monofixation syndrome. Hence if the clinician diagnoses ANAET a prompt surgical realignment of the eyes could prevent the emergence of suppression and there by amblyopia. The loss of fusion is also instrumental in the development of A or V patterns, hence a timely intervention has the potential to avert the emergence of vertical incomitance in the patients with ANAET.[18]

The possibility of an underlying lesion or malformation of the central nervous system should be kept in mind in these patients[19],[20] and careful examination to rule out divergence paralysis or bilateral 6th nerve paresis by careful motility examination, fundus examination for papilledema and optic atrophy is essential. Some clinicians may recommend routine neuroimaging in these patients but an exceedingly lower incidence of patients with underlying intracranial pathology makes a case against routine neuroimaging. We order neuroimaging only when there is an associated neurological deficit or a history of convulsion. Absence of neurological signs and presence of good fusion response with prism correction seem to be good predictors of absence of intracranial pathology.[13],[21] Nevertheless, there are now two well-documented cases of brain tumors in which good motor and sensory fusion could be shown before and after intracranial surgery. Hence presence of fusion does not rule out an intracranial pathology and the importance of a careful long-term observation should be emphasized to the parents.[21],[22],[23]

There is no definition in the literature on how late the onset for late-onset esotropia should be. We defined onset later than one year as 'late' as most of the infantile esotropes present within first six months and certainly within one year.

In our study the refractive error ranged from high myopia to high hypermetropia, there was no patient with significant anisometropia (> 1.5 diopter). Ametropia or anisometropia probably does not play any significant role in the pathogenesis of ANAET. Ahmed and Young[24] have reported ANAET in two pairs of monozygous twins and in our study one patient had the younger sister affected with congenital esotropia suggesting the hereditary basis.

The mechanism proposed by von Noorden for the pathogenesis of the Burian-Francesschetti syndrome is the loss of borderline fusion (poor fusional divergence) and the precipitation of the nonaccommodative comitant esodeviation.[13] Strabismic amblyopia associated with late-onset esotropia is perhaps the most common form of amblyopia.[25]

In our experience all the patients require surgical intervention. An early surgery could prevent the onset of amblyopia and prevent a permanent reduction in binocular vision and stereoacuity. The angle of the deviation generally remains stable. However, prism adaptation has been suggested preoperatively to bring out the full deviation and to estimate the fusional potential .[25] Use of Botulinum toxin injection in this group of patients is reported.[27] However, limitations to its use are the need of more than one injection, lack of predictability and unknown dose-response relationship.

In summary ANAET is more common than reported in the previous literature, has a variable time of onset and is associated with a high incidence of amblyopia. Timely management would avert the emergence of amblyopia and a vertical incomitance thereby promising better binocular outcome.


  Acknowledgement Top


Dr. Kulin Kothari

 
  References Top

1.
Donnelly UM, Stewart NM, Hollinger M. Prevalence and outcomes of childhood visual disorders. Ophthalmic Epidemiol 2005;12:243-50.   Back to cited text no. 1
[PUBMED]  [FULLTEXT]  
2.
Graham PA. Epidemiology of strabismus. Br J Ophthalmol 1974;58:224-31.   Back to cited text no. 2
[PUBMED]    
3.
Chew E, Remaley NA, Tamboli A, Zhao J, Podgor MJ, Klebanoff M. Risk factors for esotropia and exotropia. Arch Ophthalmol 1994;112:1349-55.  Back to cited text no. 3
[PUBMED]    
4.
Jenkins RH. Demographics: Geographical variations in the prevalence and management of exotropia. Am Orthop J 1992;42:82-7.  Back to cited text no. 4
    
5.
Yu CB, Fan DS, Wong VW, Wong CY, Lam DS. Changing patterns of strabismus: A decade of experience in Hong Kong. Br J Ophthalmol 2002;86:854-6.   Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.
Lambert SR. Are there more exotropes than esotropes in Hong Kong? Br J Ophthalmol 2002;86:835-6.   Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.
Mohney BG. Common forms of childhood esotropia. Ophthalmology 2001;108:805-9.   Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.
Legmann Simon A, Borchert M. Etiology and prognosis of acute, late-onset esotropia. Ophthalmology 1997;104:1348-52.  Back to cited text no. 8
[PUBMED]    
9.
Swan KC. Esotropia following occlusion. Arch Ophthalmol 1947;37:444.  Back to cited text no. 9
    
10.
Burian HM. Sudden onset of concomitant convergent strabismus. Am J Ophthalmol 1945;28:407.  Back to cited text no. 10
    
11.
Burian HM, Miller JE. Comitant convergent strabismus with acute onset. Am J Ophthalmol 1958;45:55-64.   Back to cited text no. 11
[PUBMED]    
12.
Franceschetti A. Acute concomitant strabismus. Ophthalmologica 1952;123:219-26.   Back to cited text no. 12
[PUBMED]    
13.
Gunter K von Noorden. Esodeviations. In : Gunter K von Noorden, Emilio Campos Editors. Theory and Management of strabismus in binocular vision and ocular motility. 5th ed. The C.V. Mosby Company: USA; 1990. p. 309.  Back to cited text no. 13
    
14.
Bielchowsky A. Das Einwartsschielen der Myopen Ber Dtsch. Ophthalmol Gws 1922;43:245.  Back to cited text no. 14
    
15.
Hoyt CS, Good WV. Acute onset concomitant esotropia: When is it a sign of serious neurological disease? Br J Ophthalmol 1995;79:498-501.  Back to cited text no. 15
[PUBMED]    
16.
Ohtsuki H, Hasebe S, Kobashi R, Okano M, Furuse T. Critical period for restoration of normal stereoacuity in acute-onset comitant esotropia. Am J Ophthalmol 1994;118:502-8.   Back to cited text no. 16
[PUBMED]    
17.
Spierer A. Acute concomitant esotropia of adulthood. Ophthalmology 2003;110:1053-6.   Back to cited text no. 17
[PUBMED]  [FULLTEXT]  
18.
Miller MM, Guyton DL. Loss of fusion and the development of A or V patterns. J Pediatr Ophthalmol Strabismus 1994;31:220-4.  Back to cited text no. 18
[PUBMED]    
19.
Anderson WD, Lubow M. Astrocytoma of the corpus collosum presenting with acute comitant esotropia. Am J Ophthalmol 1970;69:594-8.   Back to cited text no. 19
[PUBMED]    
20.
Bixenman WW, Laguna JF. Acquired esotropia as initial manifestation of Arnold-Chiari malformation. J Pediatr Ophthalmol Strabismus 1987;24:83-6.  Back to cited text no. 20
[PUBMED]    
21.
Hoyt CS. Discussion: Etiology and prognosis of acute, late-onset esotropia. Ophthalmology 1997;104:52.   Back to cited text no. 21
    
22.
Macpherson H, DeBecker I, McNeil JR. Beware: Armed and dangerous-acquired non-accommodative esotropia. Am Orthop J 1996;46:44-56.   Back to cited text no. 22
    
23.
Astle WF, Miller SJ. Acute comitant esotropia: A sign of intracranial disease. Can J Ophthalmol 1994;29:151-4.   Back to cited text no. 23
[PUBMED]    
24.
Ahmed S, Young JD. Late onset esotropia in monozygous twins. Br J Ophthalmol 1993;77:189-91.   Back to cited text no. 24
[PUBMED]    
25.
von Noorden GK. Factors involved in the production of amblyopia. Br J Ophthalmol 1974;58:158-64.  Back to cited text no. 25
[PUBMED]    
26.
Efficacy of prism adaptation in the surgical management of acquired esotropia. Prism Adaptation Study Research Group. Arch Ophthalmol 1990;108:1248-56.   Back to cited text no. 26
[PUBMED]    
27.
Dawson EL, Marshman WE, Adams GG. The role of botulinum toxin A in acute-onset esotropia. Ophthalmology 1999;106:1727-30.  Back to cited text no. 27
[PUBMED]  [FULLTEXT]  



 
 
    Tables

  [Table - 1]


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