Changes in the number and characteristics of patients with acute acquired concomitant esotropia over time: An 8-year retrospective study

1. Introduction

Acute acquired concomitant esotropia (AACE) is a distinctive type of strabismus characterized by the acute onset of concomitant esotropia with diplopia.[1] Prior published literature has provided details on classifications based on hypothetical mechanisms, the role of neuroimaging, and surgical outcomes.[2–7] AACE without intracranial lesions may not be a life-threatening condition. However, esotropia and diplopia cause discomfort and anxiety and can decrease the quality of life in patients with AACE.

Social environmental changes caused by new technologies usually affect the development and characteristics of the disease. For example, ocular and systemic problems associated with the use of digital display devices (smartphones and tablet computers) have been extensively reported in the literature.[8] Ocular problems include blurred vision, dry eye, visual fatigue, and refractive changes toward myopia.[8] Recently, an association between smartphone use and AACE onset has been reported.[9] In recent years, the coronavirus 2019 (COVID-19) pandemic has dramatically changed our lifestyles. Therefore, we investigated the clinical characteristics of patients with AACE and changes in the incidence of AACE over time.

2. Materials and methods

This retrospective study enrolled patients with AACE who visited our clinic between July 2014 and June 2022. AACE was defined as a sudden onset of concomitant (deviation in all directions of gaze differing by ≤ 5 prism diopters [PD]) non-accommodative esotropia with diplopia. This study included patients who did not have any neurological diseases associated with esotropia. Patients with any cause of fusion interruption or a history of systemic disease associated with esotropia were excluded.

The clinical characteristics of the patients with and without surgical treatment were compared to evaluate clinical factors associated with surgical treatment. Patients with ocular deviation ≥ 30 PD were considered to have large ocular deviations. The total study period (from 2014 quarters of the year 3 to 2022 Q2) was divided into 4 groups according to chronological order for evaluating the change of clinical characteristics over time. Each group of 2 years consisted of 8 quarters. The number and clinical characteristics of patients who visited the clinic and underwent surgery were compared between the 4 groups within the study period. The characteristics of patients who underwent surgery were compared among each group.

2.1. Clinical evaluation of patients with AACE

All patients underwent a full ophthalmologic examination during their initial visit. This included visual acuity and ocular alignment status tests, slit-lamp bio microscopy, refraction and fundus examinations, and stereoacuity tests. Onset of esotropia and diplopia was assessed based on the patients or parents reports. The angle of deviation was measured using an alternate prism cover test at 6-m and 33-cm fixation at every visit. A positive number represents esodeviation whereas a negative number represents exodeviation. Additionally, duction limitations were evaluated. Stereoacuity measurements were performed using the Lang I (Lang-Stereotest AG, Küsnacht, Switzerland) and Stereo Fly Stereotest (Stereo Optical Co., Chicago, IL). Neurological workups, including magnetic resonance imaging of the brain with the orbit, were performed to exclude neurological causes of esotropia in all patients. Serologic examinations, including a thyroid function test and an anti-acetylcholine receptor antibody test, were conducted to evaluate the cause of esotropia. A monocular patch or prescription of base-out prism glasses was given to relieve the diplopia. The amount of prism prescribed depended on the elimination of diplopia during distant fixation. Reduction of excessive near work, including the use of smartphones or tablet computers, was recommended during follow-up. Surgery was performed when the amount of esodeviation was confirmed to be consistent over a follow-up period of at least 4 months. All surgeries were performed under general anesthesia by a single surgeon. The forced duction test was performed under general anesthesia in all patients. Conventional bilateral medial rectus muscle recession (BMR) or unilateral medial rectus muscle recession and lateral rectus muscle resection procedures (R&R) for esotropia were performed using the surgical dose at the authors clinic (Table 1). The operated muscles were reattached directly to the sclera using 6-0 Vicryl sutures in all patients. The angle of deviation measured on the day of surgery or 1 day after surgery was defined as the immediate postoperative deviation. Postoperative follow-up visits were conducted at 1 week, 1 month, 3 months, and every 6 months thereafter.

Table 1 - Surgical dosage of bilateral medial rectus recession (BMR) and unilateral medial rectus recession and lateral rectus resection (R&R) for patients with acute acquired concomitant esotropia. Prism diopters Recession amount of BMR (millimeter) R&R Recession amounts of MR (millimeter) Resection amounts of LR (millimeter) 20 4.0/4.0 - - 25 4.5/4.5 - - 30 - 5.0 4.0 35 - 5.0 5.0 40 - 6.0 6.0 45 - 6.0 7.0 50 - 6.0 8.5 55 - 6.5 9.5

BMR = bilateral medial rectus muscle recession, LR = Lateral rectus muscle, MR = medial rectus muscle, R&R = medial rectus muscle recession and lateral rectus muscle resection.


2.2. Statistical analysis

Continuous data are presented as mean ± standard deviation, and categorical data are presented as counts and percentages. The data were analyzed using IBM SPSS Statistics ver. 20.0 (IBM Corp., Armonk, NY). Linear-by-linear analysis was employed to identify trends over time. This study was performed in accordance with the tenets of the Declaration of Helsinki and approved by the Institutional Review Board of Yeungnam University Hospital (2022-04-016). The requirement for informed consent was waived by the board due to the retrospective nature of this study and the use of anonymized patient data.

3. Results 3.1. Basic characteristics of included patients with AACE

A total of 97 patients (43 females) with AACE were included in this study. The basic characteristics of the patients are presented in Table 2. The mean age at the initial visit was 29.5 years (range: 7–73). The mean interval between the onset of diplopia and the initial visit was 25.4 months (89/97, range: 0.1–132). The mean ocular deviation at the initial visit was 22.5 ± 10.3 PD (4–60) at distance and 22.6 ± 12.3 PD at near (0–70). Mean spherical equivalent refractive error was −3.28 ± 2.96 D (+2.50 to −13.63) in the right eye and −3.13 ± 2.80 D (+2.38 to −12.88) in the left eye.

Table 2 - Basic characteristics of included patients with acute acquired concomitant esotropia. (N = 97) Sex (male: female) 54: 43 Pediatric: adult 30: 67 Mean age of initial visit (range), yr 29.5 ± 17.0 (7–73) Mean interval between onset and initial visit (range), mo 25.4 ± 29.6 (89/97, 0.1–132) Underlying disease  DM 1  HTN 5  DM & HTN 1 Initial amount of ocular deviation (range), PD  Distance 22.5 ± 10.3 (4–60)  Near 22.6 ± 12.3 (0–70) Spherical equivalent refractive errors (range), D  Right eye −3.28 ± 2.96 (+2.50 to −13.63)  Left eye −3.13 ± 2.80 (+2.38 to −12.88) Results of stereotest  Lang I test, passed (%) 34/64 (53.1)  Stereo fly stereotest (≤100 arcsec, %) 33/63 (52.4)

Arcsec = arcsecond, D = diopters, PD = prism diopters.


3.2. Comparison of clinical characteristics between patients with and without surgical treatment

During the follow-up period, 32 patients were prescribed prism glasses for relief of diplopia, with 22 (68.6%, 22/32) successfully adapting to the prism glasses. Sixty-five patients (67.0%, 65/97) underwent surgical treatment. A comparison of clinical characteristics between patients with and without surgical treatment is shown in Table 3. The mean age at the initial visit was lower for patients who underwent surgery than for those who did not (P = .005). The mean ocular deviation and the number of patients with large ocular deviation were larger in patients who underwent surgery than in those who did not (P < .001). Patients who did not undergo surgery showed better stereoacuity than those who did. Eight patients (8.2%, 8/97) showed ocular deviation < 20 PD at the initial visit, but gradually deteriorated and required surgical treatment. Mean age at surgery was 27.0 (range: 7–72) years. Mean preoperative ocular deviation was 33.5 PD (range: 20–55) at distance and 33.4 PD (range: 20–55) at near. Twenty-two patients underwent BMR and 43 underwent R&R. Mean immediate postoperative ocular deviation was 0.1 PD (−4 to 6) at distance and 0.1 PD (−6 to 8) at near. All included patients showed improvement in diplopia after surgery. Mean ocular deviation at the last visit was 1.2 PD (0–25) at distance and 0.7 PD (−4 to 20) at near over mean of 13.3 months of postoperative follow-up. Two patients who underwent BMR experienced recurrence to more than 10 PD of esotropia during follow-up.

Table 3 - Comparison of clinical factors between patients with and without surgery. Without surgery (N = 32) With surgery (N = 65) P value Sex (male: female) 15: 17 39: 26 .221 Pediatric: adult 6: 26 24: 41 .069 Mean age of initial visit (range), yr 36.3 ± 17.7 26.2 ± 15.8 .005 Mean interval between onset and initial visit (range), mo 19.5 ± 21.8 (30/32) 28.5 ± 32.7 (57/65) .178 Initial amount of ocular deviation (range), PD  Distance 14.31 ± 7.89 26.52 ± 8.80 <.001  Near 13.13 ± 9.90 27.28 ± 10.65 <.001  Patients with large ocular deviation (≥30 PD) at initial visit, n (%) 1 (3.1) 29 (44.6) <.001 Spherical equivalent refractive errors (range), D  Right eye −2.76 ± 2.72 −3.53 ± 3.06 .229  Left eye −2.70 ± 2.51 −3.34 ± 2.92 .294 Results of stereotest  Lang I test, passed (%) 6/8 (75.0) 28/56 (50.0) .185  Stereo fly stereotest (≤100 arcsec, %) 7/8 (87.5) 26/55 (47.3) .056

arcsec = arcsecond, D = diopters, PD = prism diopters.


3.3. Changes in numbers and characteristics of patients with AACE over time

The number of patients who initially visited the clinic and underwent surgical treatment from 2014 Q3 to 2022 Q2 is shown in Figure 1. The number of patients who initially visited the clinic increased during the study period. The number of patients who underwent surgery decreased during the period from 2018 Q3 to 2020 Q2 when compared with 2016 Q3 to 2018 Q2. However, the number of patients who underwent surgery increased and was the highest from 2020 Q3 to 2022 Q2. The clinical characteristics of the patients who underwent surgery in each period are shown in Table 4. The mean age at the initial visit continuously decreased throughout the study period; however, it failed to reach statistical significance (P = .155, linear-by-linear association). The percentage of patients with large ocular deviation increased over time among patients with AACE who underwent surgical treatment (Fig. 1, P = .037, linear-by-linear association). This means that there was an increasing trend in patients with large ocular deviation over time. Other clinical factors showed no significant differences between periods.

Table 4 - The clinical characteristic of patients who underwent surgery treatment among each period. 2014 Q3–2016 Q2 2016 Q3–2018 Q2 2018 Q3–2020 Q2 2020 Q3–2022 Q2 P value Number of patients 3 21 12 29 - Sex (male: female) 2:1 13:8 7:5 17:12 .757 Pediatric: adult 1:2 6:15 5:7 12:17 .394 Mean age of initial visit, yr 35.0 ± 24.8 31.5 ± 19.4 23.3 ± 13.1 22.6 ± 11.9 .155 Preoperative ocular deviation, PD  Distance 31.67 ± 7.64 32.14 ± 9.95 35.00 ± 11.48 34.14 ± 8.14 .804  Near 30.00 ± 5.00 30.71 ± 9.39 35.83 ± 10.62 34.83 ± 8.40 .290 Patients with larger ocular deviation (≥ 30 PD), n (%) 2 (66.7) 11 (52.4) 8 (66.7) 24 (82.8) .037 Spherical equivalent refractive errors. D  Right −1.29 ± 2.53 −2.96 ± 2.48 −3.57 ± 2.60 −4.17 ± 3.57 .313  Left −1.13 ± 2.76 −2.82 ± 2.65 −3.75 ± 2.33 −3.84 ± 3.29 .353

D = diopters, PD = prism diopters, Q = quarters.


F1Figure 1.:

The changes of patient with acute acquired concomitant esotropia (AACE) from 2014 quarters of the year (Q)3 to 2022 Q2. The number of patients who initially visited clinic increased throughout the study period. The number of patients who underwent surgery decreased during the period from 2018 Q3 to 2020 Q2 when compared with 2016 Q3 to 2018 Q2. However, the number of patients who underwent surgery increased and was the highest from 2020 Q3 to 2022 Q2. The percentage of patients with large ocular deviation (≥30 PD) from the 2014 Q3 to 2022 Q2. The percentage of patients with large ocular deviation increased over time among patients with AACE who underwent surgical treatment (P = .037, linear-by-linear association). This means that there was an increasing trend in patients with large ocular deviation over time. PD = prism diopters.

4. Discussion

The number of patients with AACE has increased over the years. There was an increasing trend in patients with large ocular deviation (≥30 PD) over time among patients with AACE who underwent surgical treatment. The patients who underwent surgical treatment showed more ocular deviation and were younger at the initial visit than in patients who did not undergo surgical treatment.

AACE is divided into 3 distinct types based on hypothetical causes and refractive errors.[1] Type I (Swan type) occurs following disruption of fusion by monocular occlusion. Type II (Franceschetti type) has no apparent underlying cause other than debilitating illness or physical or emotional stress. Type III (the Bielschowsky type) is associated with myopia. Treatment of esotropia and diplopia is the primary focus of patients with AACE without neurological diseases. Traditionally, AACE is considered to be a relatively uncommon type of strabismus. However, the incidence of AACE has recently increased.[10]

A study by Zhu et al[10] showed a marked increase in the number and proportion of surgical patients with AACE in recent years and also demonstrated that young adults account for the majority of individuals affected by AACE. However, previous studies have not evaluated changes in the number and clinical characteristics of patients with AACE over time. We investigated the incidence and characteristics of patients with AACE over the last 8 years.

The number of patients with AACE increased over time during study period. There was increasing trends of patients with large ocular deviation over time among patients with AACE who underwent surgical treatment. It is well known that an increase in excessive close visual activity, such as smartphone use, is associated with the onset of AACE.[9–11] Excessive near work and a naturally stronger convergence can lead to an imbalance between accommodation and vergence, resulting in dynamic activation of the medial rectus muscles with or without divergence weakness, thus producing esotropia.[9,12] Since smartphones were first introduced to the market, their screen size has increased from the 2010s onward.[13] Large screen size is likely to lead to higher smartphone adoption by simultaneously promoting both the utilitarian and hedonic qualities of smartphones.[13] These market changes may also increase smartphone use, resulting in increased development of AACE.

Interestingly, the number of patients who underwent surgery decreased from 2018 Q3 to 2020 Q2 and then increased again in recent years. Lee et al first pointed out in 2016 that excessive smartphone use might influence AACE development in adolescents.[9] Based on their report, we recommended decreasing the use of smartphones in patients with AACE. This might have led to a decrease in the number of patients who underwent surgery from 2018 Q3 to 2020 Q2. However, 2020 Q1 was the onset of the COVID-19 pandemic. COVID-19 lockdown led to important lifestyle changes, including restriction of external daily activities and increased use of smartphones.[14] These changes are likely to have a significant impact on the development and worsening of myopia.[15] Similarly, there have been reports of AACE development during the COVID-19 pandemic.[12,16] A study by Mohan et al showed prolonged near work during smartphone use for e-learning might lead to the development of AACE in children.[17] The COVID-19 pandemic may result in another surge in the number of patients undergoing surgery for AACE.

Patients who underwent surgical treatment showed more ocular deviation and were younger at the initial visit compared with patients who did not undergo surgical treatment. Although the difference did not reach statistical significance, the mean age of patients with AACE who underwent surgery decreased throughout the study period. This may be because younger patients are more susceptible to exposure to digital devices.[18] In this study, 8 patients showed ocular deviation lower than 20 PD at the initial visit, but gradually deteriorated and required surgical treatment. Half of these were pediatric patients with AACE. Younger individuals use smartphones longer, and their usage is directed towards entertainment and social interactions compared with older individuals.[19] Pediatric patients may be more vulnerable to excessive smartphone exposure.[14,18] Strict restrictions on smartphone use are required in pediatric patients with AACE.

Different therapeutic approaches can be considered in patients with AACE. Prism glasses can be prescribed to patients with relatively small or moderate amounts of ocular deviation.[1] Approximately 2-thirds of patients successfully adapted prism glasses for the relief of diplopia in this study. Wu et al[20] suggested a step-by-step prismatic treatment in patients with an AACE of 25 PD or less. Surgical treatment is recommended for patients with AACE with a large ocular deviation. BMR and unilateral R&R are the most common surgical procedures performed in patients with AACE.[3,5,6] The surgical outcomes of AACE are relatively favorable compared with those of other types of esotropia.[5] In this study, all patients who underwent surgery showed an improvement in diplopia.

This study has some limitations. First, this study analyzed the development of patients with AACE at a single institution. Therefore, there are limitations in generalizing the prevalence of AACE in the entire population. Further studies covering multiple centers are required. Second, the patterns of individual smartphone use, including screen time and device screen size, were not obtained in this study. Previous studies have usually measured this information based on the patient’s self-reporting. Recently, some smartphones provided real-time reports showing how much time and what contents users used on their devices.[21] Further studies using these features will help understand the association between smartphone use and AACE onset.

Author contributions

Conceptualization: Chae Won Lim, Won-Jae Kim.

Data curation: Chae Won Lim, Won-Jae Kim.

Formal analysis: Chae Won Lim, Jinwoo Lee, Won-Jae Kim.

Funding acquisition: Won-Jae Kim.

Investigation: Chae Won Lim, Jinwoo Lee, Won-Jae Kim.

Methodology: Chae Won Lim, Won-Jae Kim.

Project administration: Won-Jae Kim.

Resources: Won-Jae Kim.

Supervision: Jinwoo Lee, Won-Jae Kim.

Validation: Won-Jae Kim.

Visualization: Chae Won Lim, Won-Jae Kim.

Writing – original draft: Chae Won Lim, Jinwoo Lee, Won-Jae Kim.

Writing – review & editing: Chae Won Lim, Jinwoo Lee, Won-Jae Kim.

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