Original Research

Results of Surgery for Congenital Esotropia

Pedro D. Castro MD, Alina Pedroso MD, Lourdes Hernndez MD, Rosa M. Naranjo MD, Teresita de Jess Mndez MD,
Alejandro Arias MD

ABSTRACT
INTRODUCTION Congenital esotropia is a convergent deviation of
the eyes when fixating on an object. It appears during the first six
months of life and affects muscle structure and physiology, as well as
the relation of the eyes with the environment, retinal relations and neural integration phenomena. Amblyopia develops in 3050% of those
affected. Timely surgery is effective in most cases.
OBJECTIVE Describe surgical results in congenital esotropia cases
treated in the Pediatric Ophthalmology and Strabismus Service of the
Ramon Pando Ferrer Ophthalmology Institute in Havana between
January 2008 and May 2009.
METHODS A descriptive, retrospective study was carried out through
review of 127 cases (aged 118 years, 55 male and 72 female) operated on for congenital esotropia between January 2008 and May
2009. Variables used were: age at surgery, degree of preoperative
amblyopia, type of fixation, refractive error, associated conditions, type
of surgery performed and pre- and postoperative deviation angles.

INTRODUCTION
Strabismus is an ophthalmological disorder in which the eyes are
misaligned; it affects 24% of children.[1] About half of these disorders are esodeviations, whose causes are anatomical, neurological, mechanical, refractive, genetic and accommodative.[2,3]
Congenital esotropia is a convergent deviation of the visual axes
when fixating on an object. It appears in the first six months of
life, generally between two and four months. While timing of its
first clinical signs varies, it is the result of a congenital failure to
develop normal binocular vision, probably genetically-determined
development in normal binocular vision.[4,5] In general, it occurs
in children who show orthotropia or exodeviations at birth. Congenital esotropia should not be confused with other true congenital deviations (occasionally fixed esodeviations), which can be
detected from the first day of life and are clinically well defined.
Reported prevalence of the condition varies. Some authors say it
affects some 0.1% of the population;[6] Mohney et al. reported a
9year period prevalence of 0.27%.[1] Prevalences between 0.67
and 2% have been found among ophthalmological patients;[7,8]
and a study of strabismus patients in Mexico found congenital
esotropia to be the most frequent type (28.46%), with similar rates
for both sexes.[911] There are no prevalence data on this condition in Cuba.
In most cases, congenital esotropia appears to be caused by defective development of fusion (cerebral integration of images perceived by both eyes). This hypothesis is supported by some patients failure to achieve normal binocularity even with very early
alignment of the eyes. However, environmental factors cannot be
discounted, since a high frequency of strabismus and amblyopia is notable among low birth-weight and premature infants, and
those suffering perinatal hypoxia. Smoking, drugs and alcohol
abuse during pregnancy also disrupt brain development and are
closely associated with amblyopia and strabismus.[12] Addition18

Peer Reviewed

RESULTS The largest number of congenital esotropia cases were

found in children <2 years old (38.6%). Before surgery, 37% of cases
studied showed mild amblyopia; 22.8% moderate and 12.6% severe;
27.6% did not cooperate with visual acuity testing. With respect to
fixation: 91.3% showed central fixation and 8.7% eccentric. Refractive errors found were: mild hyperopia (65.4%), moderate hyperopia
(29.1%), and myopia (5.5%). Associated conditions were: overaction of the inferior oblique muscles (48%), latent nystagmus (16.5%),
and dissociated vertical deviation (8.7%); no alterations were found
in 26.8% of cases. The most frequently used surgical treatment was
bilateral medial rectus muscle recession (92.1%). The most common
preoperative deviation angle range was 3140 prism diopters (PD) in
51.9% of patients; the postoperative deviation angle most commonly
found after 12 months was <10 PD in 64.6% (orthotropia).
CONCLUSIONS Surgery, most commonly with medial rectus muscle
resection, was effective in correcting congenital esotropia.
KEYWORDS Esotropia/congenital; esotropia therapy; oculomotor muscles/surgery; strabismus; squint; amblyopia; vision, binocular; Cuba

ally, inheritance may play a role in the pathogenesis of congenital

esotropia; some authors report binocular vision anomalies in 16%
of parents of children with congenital esotropia.[12,13]
Congenital esotropia may be accompanied by amblyopia (reduction in uni- or bilateral best corrected visual acuity not attributable
to structural abnormality of the eye or posterior visual pathway),
thus establishing the sensory nature of the impairment.[14] Ocular misalignment is in fact the main cause of amblyopia. Constant
esotropic deviations, rather than alternating ones, are the fundamental cause of strabismic amblyopia.[15] Amblyopia is classified according to best corrected visual acuity (VA): mild (0.90.6),
moderate (0.50.3) and severe (0.20.05).[15] Some authors do
not explicitly define mild amblyopia and others ignore it, referring
only to moderate and severe amblyopia.[15]
Limitations in social and psychological development resulting
from amblyopia make effective and early correction imperative.
Delay in surgical alignment of congenital esotropia is an important risk factor for postoperative amblyopia.[5,12] However, criteria vary regarding the optimal timing of surgery. Costenbadera
recognized authority in strabismus managementsuggested surgery was needed at 12 months to expect functional cure.[4] Most
ophthalmologists agree that surgery should be done early, aligning the eyes before 24 months of age to attain binocular vision.
Some hold that surgery may be performed in healthy children as
early as 46 months of age to maximize binocular function and
improve stereopsis.[11,12]
Significant refractive errors should be corrected and treatment of
amblyopia undertaken before surgery, creating better visual conditions, and mitigating accommodation or anisometric defects. In
very farsighted children with a small deviation angle <20 prism
diopters (PD), correcting the refractive error may avoid surgery.
When amblyopia is suspected, occlusion therapy is indicated
MEDICC Review, January 2011, Vol 13, No 1

This article has been corrected. The specific correction appears at the end of this document.

Original Research
daily patching time dependent on the childs ageto force the
weaker eye to fixate.[12] Patching is not just ideal to prevent or
treat amblyopia but also to improve esotropia as such; by reducing hypertonicity in the medial rectus muscle and stimulating
abduction of the non-occluded eye, it enhances binocular vision
from a sensory perspective.[13]
Conventional operations routinely used for other clinical forms
of esotropia are often inadequate for alignment in cases of congenital esotropia, the latter sometimes involving large deviation
angles; nor are these surgeries generally effective for correcting
torticollis.[12] Hence specific surgical procedures are needed.
Bilateral recession of the medial rectus muscles was already being done in the 1950s. Costenbader posited that congenital esotropia treatment should be based on weakening the medial rectus
muscles to prevent eventual loss of cortical inhibition of hyperactive convergence.[4,5,12,14] This is the rationale for standard
treatment: bilateral recession of the medial rectus muscles. The
extent of recession is generally based on the degree of deviation
with near fixation, since in infants it is difficult to obtain exact measurements with distant fixation. In older patients with significant
irreversible amblyopia, surgery should be limited to the amblyopic
eye, with recession of the medial rectus muscle and resection of
the lateral rectus muscle.[4,5,12,14]
In Cubas national health system, primary health care is key to
identifying children with congenital esotropia. They are seen in
well-baby visits involving a neighborhood family doctor and pediatrician, and later referred to ophthalmology and strabismus
services in provincial pediatric hospitals throughout the country,
including six in Havana plus the Ramn Pando Ferrer Ophthalmology Institute (ICO, its Spanish acronym).
School-age children who have not received early surgery are
enrolled in public special education schools, where the goal is to
develop each childs visual function as much as possible, optically and perceptually, through a comprehensive system of education and rehabilitation. This involves methodologies based on
physiological, ophthalmological, psychological and pedagogical
principles to faster greater independence and active social participation.[16] In the case of amblyopic children, they are able
to follow their daily patching routine with fewer psychological
implications, especially as they reach ages when appearance
becomes more important. Parents receive guidance on how to
manage these conditions and on the rehabilitation necessary for
their childs eventual integration into the mainstream educational
system.[16]
The ICO is Cubas national ophthalmology reference center.[17]
The ICO Pediatric and Strabismus Service receives patients referred from Havana and across the country for treatment intended
to achieve ocular alignment, correct torticollis, and prevent amblyopia, with a minimum of surgical interventions. It also provides
psychological support for the children and their families. In addition, the Service receives those congenital esotropia patients
whose comorbidities (dissociated vertical deviations, overaction
of the oblique muscles, vertical anisometropia) complicate their
surgical management by other hospitals around the country.

by the childs age. Refractive errors are corrected to improve vision as much as possible and to mitigate associated accommodation factors or anisometropia. When a patient has alternating
esotropia from onset, with a deviation angle >20 PD and without
significant neurological damage, surgery is performed prior to 24
months to ensure the best chance for normal binocular vision development.
The objective of the present study is to describe surgical results
in congenital esotropia cases treated at the Ramon Pando Ferrer
Ophthalmology Institute in Havana between January 2008 and
May 2009.

METHODS
A descriptive retrospective study was designed, based on review
of clinical records, to evaluate results of surgical treatment of congenital esotropia in cases referred to the ICO Pediatric and Strabismus Service from January 2008 through May 2009.
Of 149 records reviewed of patients aged <18 years, 127 met inclusion criteriadeviation angle >20 PD and no significant neurological damage. Of those, 55 were male and 72 female. Excluded
were those whose congenital esotropia did not fulfill surgical criteria (i.e. deviation angle 20 PD or significant neurological damage), or whose clinical records were incomplete.
Variables studied were: age at surgery, sex, degree of preoperative amblyopia, type of fixation, refractive error, comorbidities,
type of surgery performed and pre- and postoperative deviation
angles (Table 1).
Table 1: Definition of variables
Variable

Age at surgery (years)

Degree of amblyopia
(visual acuity, VA)
Type of fixation

Refractive error
(diopters, D)

Associated conditions

Deviation angle
(prism diopters, PD)

Type of surgery

Our prophylactic or therapeutic practice in suspected amblyopia is

alternating patching before age one, with the regimen determined
MEDICC Review, January 2011, Vol 13, No 1

Peer Reviewed

Parameters
<2
24
57
810
>10
(0.60.9)
(0.20.5)
(0.050.2)

Mild
Moderate
Severe
Central
Eccentric
Mild hyperopia
<3.00
Moderate hyperopia
36
High hyperopia
>6
Myopia
(same as for hyperopia)
Astigmatism
Overaction of inferior oblique
muscles
Dissociated vertical deviation
Torticollis
Nistagmus
<10
1020
2130
3140
4150
>50
Recession of both medial rectus
muscles
Monocular surgery (recession
medial rectus muscle, resection
lateral rectus muscle)

19

Original Research
Techniques and procedures Each cases clinical record was
reviewed to confirm that the following procedures had been carried out:
General ophthalmological examination: date, duration of deviation, age at onset, associated symptoms and ocular examination of both eyes; slit-lamp examination of the anterior segment
(cornea, iris, pupil and lens);
Ocular motility examination: deviation angle in primary-gaze
position with Hirschberg method; cover test; ocular movements
(ductions and versions); uncorrected and best corrected VA;
ocular dominance test in preverbal children aged <2 years; Kay
Picture test for children 25 years; and Snellen E test and letter
and number charts for those >5 years;[18]
Motor function study, determining deviation angle with distance
and near prisms, using the Krimsky method if VA by prism cover test 0.4; or, modified Krimsky if VA<0.4 or children did not
cooperate during the exam;[18]
Study of sensory function based on uncorrected and best corrected VA;
Dilation of both pupils three times with 1% cyclopentolate, at
5-minute intervals; after 60 minutes, with paralyzed accommodation to define type of fixation and inspect fundus with binocular indirect ophthalmoscope; and
Surgical treatment for bilateral symmetric cases according to
the American Academy of Ophthalmology guidelines, which
describe extent of medial rectus recession for given deviation
angles; for unilateral cases, monocular recession-resection, selecting appropriate muscle length.[14]
Ethical considerations The study was approved by the ICO Ethics Committee. Data were obtained exclusively from clinical records, maintaining patient anonymity.

The most frequent comorbidity was overaction of the inferior

oblique muscles (48%); the least frequent was dissociated vertical deviation (8.7%). No comorbidities were found in 26.8% of
cases (Figure 2).
Figure 2: Comorbidities observed (n=127)

Dissociated
vertical deviation

Latent
nystagmus

Overaction of
inferior oblique
muscles

No
comorbidities

10

20

30

40

50
%

The most common surgery performed was recession of both medial rectus muscles (92.1%), while monocular surgery (recession
of the medial rectus and resection of the lateral rectus) was performed in only 7.9% of cases.

Data analysis Primary data were processed using Excel 2003.

Qualitative variables were statistically described using absolute
and relative frequencies, percentages calculated for the latter.
Data were organized in tables and figures.

The most frequent preoperative deviation-angle range was 3140

PD in 66 cases (51.9%). One year after surgery, 64.6 % of cases
were orthotropic (Table 2).

RESULTS

Clinical records showed most patients received early medical

attention, providing opportunity for appropriate management.
Hence, the majority of cases in the present study were under 2
years old. Beginning occlusive treatment at 6 months of age and
correcting refractive errors promotes the best possible visual conditions for surgery before 24 months.

The largest group of congenital esotropia cases referred to the

ICO for evaluation consisted of children aged <2 years (38.6% of
total) (Figure 1).
Before surgery, 37% of cases presented mild amblyopia; 22.8%
moderate; 12.6 % severe; and 27.6% did not cooperate in VA testing (children <3 years old). By far the most common type of fixation was central (91.3%); 8.7% had eccentric fixation. The most
frequent refractive error was mild hyperopia (65.4%), followed by
moderate hyperopia (29.1%) and myopia (5.5%).
Figure 1: Surgical patients by age (n=127)
9.4%

<2

38.6%

8.7%

2-4
5-7
8-10
>10

26.8%
16.5%

20

Age in years

Peer Reviewed

DISCUSSION

While authors still disagree on optimal age for surgery, Prieto Daz and others suggest that the earlier ocular alignment is
achieved, the better the functional result attained.[12] He studied
171 surgically-aligned cases aged >4 years. Of these, 123 were
selected because they fully cooperated: 108 (87.8%) showed fusion (assessed using Bagolini striated lenses); and 92 (74.8%)
a degree of stereopsis (assessed by Titmus test). He observed
close association between fusion and stereopsis and age at alignment: all 76 cases that were aligned between 8 and 17 months of
age showed fusion and stereopsis, while of the 16 aligned after
36 months of age, only 8 showed fusion and none showed stereopsis.
There is a period of sensory plasticity in the first 18 months of
life during which subnormal or anomalous binocular vision may
still develop fully, the potential gradually decreasing thereafter
until around 36 months.[12] Helveston held that 4 months old
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Original Research
Table 2: Pre- and 12-month postoperative deviation angle by age (n=127)
Age Group
(Years)

Surgical
Status

Deviation angle (prism diopters)

Pre
Post
Pre
Post
Pre
Post
Pre
Post
Pre
Post

32

12

23

65.3

57.1

67.7

72.7

58.3

1020

Total

Pre

Total

Post

82

64.6

<2
24
57
810
>10

<10

18.4

33.3

17.6

18.2

33.3

2130
2
6
0
2
0
5
1
1
3
1

%
4.1
12.2
0.0
9.5
0.0
14.7
9.1
9.1
25.0
8.3

3140
24
2
10
0
20
0
6
0
6
0

%
49.0
4.1
47.7
0.0
58.8
0.0
54.5
0.0
50.0
0.0

4150
20
0
9
0
14
0
4
0
3
0

%
40.8
0.0
42.9
0.0
41.2
0.0
36.4
0.0
25.0
0.0

4.7

66

51.9

50

28

22.0

15

11.8

1.6

was the earliest age for congenital esotropia surgery, if diagnostic certainty was possible.[11] Yet, when he presented 10year follow-up results on 12 cases operated on at about that
age, he found no better results than for those who received
surgery at 12 months of age. Early alignment produces postoperative esotropia with a small deviation angle (ortho 10 PD),
or, exceptionally, orthotropia.[12] In the present study, surgical alignment was satisfactory in a high percentage of children
aged <2 years.
Preoperative, most cases presented mild amblyopia, consistent
with other reports of amblyopia in approximately 4050% of children with congenital esotropia.[19,20] This is likely due to the
presence of fixation micro-nystagmus causing visual maturation
delay, because the fovea does not receive stable images. However, this deficit usually improves gradually over time.[19]
The majority of cases (91.3%) had central or foveal fixation, corresponding to the mild degree of amblyopia found. In congenital
esotropia, patients may alternate fixation or show a fixation preference for one eye. A strong fixation preference indicates significant amblyopia and should be treated by patching the dominant
eye before strabismus surgery, as done at ICO.
Mild hyperopia was the most frequent refractive error, consistent
with the literature.[12,13,19,20] If congenital esotropia patients
have hyperopia, it tends to be moderate (around +2.50 D) and
does not influence deviation angle. According to Prieto Daz, in a
study of 256 congenital esotropia cases, only 14.4% (37 cases)
showed 2.50 D spherical hyperopia.[12] Other studies reveal
that these patients refractive errors are similar to those in the
general population and suggest that in cases where hyperopia is
>3.00 D, it should be carefully assessed, since it could result in
overcorrection after esotropia surgery.[1,3]
In this study, the ocular disorder most commonly associated with
congenital esotropia was overaction of inferior oblique muscles,
consistent with standard texts indicating that this is more frequent
in congenital cases than in those of later onset (acquired comitant
esotropia).[12,13] In a study of 120 consecutive cases of congenital esotropia, 70% showed overaction of the oblique muscles,
46.7% of the inferior obliques. Primary overaction of the inferior
oblique muscle was found in 72% of patients, sometimes upon
MEDICC Review, January 2011, Vol 13, No 1

>50
3
0
2
0
0
0
0
0
0
0

%
6.1
0.0
9.5
0.0
0.0
0.0
0.0
0.0
0.0
0..0

Total
49
49
21
21
34
34
11
11
12
12

39.4

3.9

127

0.0

0.0

127

first examination and sometimes later. Overaction of the superior

oblique muscle is less common.[12]
More recently it has been stated that in children with esotropia
and loss of fusion, normal vergence control mechanisms are lost.
The neural impulse favors extorsion over intorsion, leading to progressively increased tone in the inferior oblique muscle with secondary shortening and reduction in tone of the superior oblique
with secondary elongation, similar to what happens in sensory
esotropia.[5,21]
In the present study, the most common surgical technique was
recession of both medial rectus muscles, in line with other authors.[5,1214,19,21,22] This is the most frequently used technique, with the extent of recession determined by the deviation
angle.[5,12,14] We prefer this technique to recession-resection
because its symmetry allows angle correction without causing
incomitance on lateroversion and because it simultaneously corrects the bilateral motor abduction imbalance characteristic of
these children. All clinical phenomena (crossed fixation, abduction limitation, optokinetic nystagmus) were symmetrical and bilateral, and in a high percentage preoperative examinations are
also symmetrical (alignment test under general anesthesia 74%;
passive duction test 71%; test for muscle elongation 79%), so we
prefer to use symmetrical rather than asymmetrical surgical correction.[5,19,21]
In this study, preoperative deviation angle in most cases was in
3140 PD range, consistent with other reports.[12,1922] One
year after surgery, 64.6% of cases had achieved alignment,
similar to results obtained by Helveston.[23] Cases that maintained deviation angles of 1020 PD were considered functionally and aesthetically acceptable, not requiring a second
surgery. These showed continued spontaneous improvement
in deviation angle with corrective lenses for moderate hyperopia. All this is consistent with results of other studies assessing
evolution at 6 months to one year, reporting success rates of
7080%.[20,2224]
A pharmacological alternative to surgery now exists for congenital esotropia, used as first line treatment in children aged <18
months who do not have important vertical deviations. It involves
injecting the medial rectus muscles with botulinum toxin (Botox)
Peer Reviewed

21

Original Research
to reduce deviation angle. This is done in several sessions, generally once every three months. According to studies reviewed,
the treatment has obtained very good horizontal results.[12,13,25]
Other authors such as Helveston consider that, if general anesthesia is administered to expose the muscle for Botox injection,
the muscle could well be weakened the traditional way with only
a few more minutes of surgery. Helveston contends that this drug
has an important but limited role, given that most studies show the
need for multiple injections to maintain the effect.[23]
We have no direct experience with botulinum toxin for congenital
esotropia. At this point, we share Helvestons opinion and consider that the treatment of choice is still conventional surgery,
which results in more stable alignment in most cases and avoids
repeated invasive procedures, with the attendant risks of general
anesthesia.

A limitation of this study is that it was too short to permit assessment of visual outcomeVA, fusion, and stereopsisin relation to age at alignment. This exploration of binocularity could
be the objective of further research including more cases from
the countrys pediatric hospitals and special schools.[26] Ideally, these institutions should participate in determining prevalence of congenital esotropia in Cuba and assessing treatment
results.

CONCLUSIONS
This study of surgical treatment of congenital esotropia revealed
success rates in Cuba similar to those seen internationally, with
satisfactory rates of orthotropia one year postoperatively. Further
study involving follow-up of these and additional cases is recommended to verify the relation between age at alignment and sensory results obtained.

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Peer Reviewed

THE AUTHORS
Pedro Daniel Castro Prez (Corresponding author: pedrocastro@infomed.sld.cu),
ophthalmologist, Ramn Pando Ferrer
Ophthalmology Institute, Havana, Cuba.
Alina Pedroso Llanes, ophthalmologist,
Ramn Pando Ferrer Ophthalmology Institute, Havana, Cuba.
Lourdes Hernndez Santos, ophthalmologist, Ramn Pando Ferrer Ophthalmology Institute, Havana, Cuba.
Rosa Mara Naranjo Fernndez, ophthalmologist and assistant professor, Ramn
Pando Ferrer Ophthalmology Institute, Havana, Cuba.
Teresita de Jess Mndez Snchez, ophthalmologist and associate
professor, Ramn Pando Ferrer Ophthalmology Institute, Havana, Cuba.
Alejandro Arias Daz, ophthalmologist,
Ramn Pando Ferrer Ophthalmology Institute, Havana, Cuba.

Submitted: June 26, 2010

Approved for publication: December 31, 2010
Disclosures: None

MEDICC Review, January 2011, Vol 13, No 1

Erratum
Page 18, Abstract, Methods, first sentence should read: "A descriptive, retrospective study was carried out through
review of 127 cases (aged 118 years, 55 male and 72 female) operated on for congenital esotropia between
January 2008 and May 2009."