Surgical treatment of unilateral acquired superior oblique muscle palsy in adults by inferior oblique belly transposition: a retrospective analysis.

PURPOSE: Evaluate and analyze the efficacy of inferior oblique belly transposition (IOBT) in treating adult patients with diplopia and small-angle hypertropia caused by mild to moderate inferior oblique overaction (IOOA) secondary to acquired superior oblique palsy (SOP). METHODS: Nine adult patients with diplopia and small-angle hypertropia associated with mild to moderate IOOA secondary to unilateral acquired SOP were included in the current retrospective study. All patients received the IOBT procedure between February 2019 and May 2023 at The Second People's Hospital of Jinan and were followed up for more than 6 months after the surgery. During the procedure, the belly of the inferior oblique muscle was fixed to the sclera at 5 mm posterior to the temporal insertion of the inferior rectus muscle. The following indicators were reviewed pre- and post-surgery: the vertical deviation (VD) in the primary position and in the Bielschowsky test, the fovea disc angle (FDA) of the affected eye, changes in IOOA, and diplopia. RESULTS: After IOBT, the VD in the primary position decreased from 7.22△ ± 1.72△ (range 4△-10△) to 1.22△ ± 1.30△ (range 0△-3△). The VD in the Bielschowsky test decreased from 13.00△ ± 1.80△ to 3.22△ ± 1.09△. The FDA decreased from 10.02° ± 3.34° to 6.26° ± 1.91°. The grade of IOOA was reduced from 2.00 (1.00, 2.00) to 0.00 (0.00, 1.00). All changes were statistically significant (P < 0.001 or P = 0.006). Diplopia was resolved completely for all patients. CONCLUSIONS: IOBT can effectively treat adults with diplopia and small-angle hypertropia caused by mild to moderate IOOA secondary to acquired SOP.

The Outcome of Inferior Oblique Myectomy for Apparent Inferior Oblique Overaction Associated with Craniosynostosis.

PURPOSE: Strabismus in patients with craniosynostosis is common, but surgical correction of strabismus in these patients remains challenging. We report our findings in six patients (four of whom were Korean) with craniosynostosis who underwent strabismus surgery to specifically address V-pattern horizontal strabismus with moderate-to-severe inferior oblique (IO) overaction, using IO myectomy at a single tertiary hospital between 2005 and 2016. METHODS: We recorded preoperative characteristics including sex, age, type of strabismus, versions grading, refractive error, and visual acuity. The grading of cyclorotation of horizontal rectus muscles by V-pattern categorized using coronal computed tomography imaging. RESULTS: Of the six patients, exodeviation was found in four patients and vertical deviation in two patients in primary position. One patient had both horizontal and vertical strabismus. Available computed tomography imaging showed that V-patterns were category 1 (mild) in two patients, category 2 (moderate) in one patient, and category 3 (severe) in two patients. Complete success was defined as absence of IO overaction any more. Overall complete success rate of IO myectomy was 83.3%. CONCLUSIONS: IO myectomy appeared to have some benefits in V-pattern horizontal strabismus with moderate-to-severe IO overaction in patients with craniosynostosis.

Maternal Hypertensive Disorder in Pregnancy and Childhood Strabismus in Offspring.

Importance: Maternal hypertensive disorder in pregnancy (HDP) might affect ocular health in offspring; however, its association with strabismus remains unclear. Objective: To examine the association of maternal HDP with overall and type-specific strabismus in offspring. Design, Setting, and Participants: In the Jiangsu Birth Cohort study, a population-based study in China, pregnant women were recruited from April 24, 2014, to November 30, 2018. A total of 6195 offspring had maternal HDP diagnosis information, of whom 3078 were excluded due to having no information on ocular alignment or due to having ocular diseases other than strabismus or refractive error. Offspring underwent ocular examinations at 3 years of age, completed May 21, 2022. Data were analyzed from May 28, 2022, through December 15, 2023. Exposure: Maternal HDP, categorized into hypertension and preeclampsia or with blood pressure (BP) well controlled (systolic BP, <130; diastolic BP, <80 mm Hg) and poorly controlled (systolic BP, ≥130; diastolic BP, ≥80 mm Hg). Main Outcomes and Measures: The primary outcome was the incidence of strabismus in offspring. Poisson generalized linear mixed models were used to estimate the association between maternal HDP and strabismus. Results: Among the included 3117 children (mean [SD] age, 36.30 [0.74] months; 1629 boys [52.3%]), 143 (4.6%) were exposed to maternal HDP and 368 (11.8%) had strabismus. Offspring exposed to maternal HDP had an 82% increased risk of overall strabismus (relative risk [RR], 1.82 [95% CI, 1.21-2.74]), an 82% increased risk of exophoria (RR, 1.82 [95% CI, 1.11-3.00]), and a 136% increased risk of intermittent exotropia (RR, 2.36 [95% CI, 1.13-4.93]) compared with unexposed offspring. When considering the type of maternal HDP, the risk for all strabismus was high for offspring exposed to preeclampsia (RR, 2.38 [95% CI, 1.39-4.09]) compared with unexposed offspring. When considering the BP control level of maternal HDP, the risk for all strabismus was high for offspring born to mothers with HDP and poorly controlled BP (RR, 2.07 [95% CI, 1.32-3.24]) compared with unexposed offspring. Conclusions and Relevance: These findings suggest that maternal HDP is associated with an increased risk of offspring strabismus. Early screening of strabismus might be recommended for offspring with maternal HDP. Further exploration of the underlying mechanism of the association between HDP and strabismus is warranted.

Anterior segment optical coherence tomography (AS-OCT) in strabismus following trauma.

We describe 3 cases of ocular trauma that presented with strabismus. Anterior segment optical coherence tomography was used to delineate the morphology of extraocular muscle insertions and proved helpful in diagnosis and management.

Assessment of surgical strategies for management of complicated strabismus reoperation in Graves' ophthalmopathy.

PURPOSE: Strabismus reoperation in Graves' ophthalmopathy (GO) is complicated and challenging. The purpose of this study was to evaluate the various surgical strategies of strabismus reoperation and their outcomes in patients with GO. METHODS: A retrospective study was conducted on strabismus reoperations performed at the Zhongshan Ophthalmic Center of Sun Yat-sen University, Guangzhou, China from 2008 to 2018. Data collected included sex, age at surgery, duration of deviation, ocular alignment, ocular motility, various surgical procedures performed and surgical outcomes. Surgical methods included rectus recession for newly developed strabismus, rectus resection for undercorrection and anterior advancement of a previously recessed rectus for overcorrection. Surgical success was defined as an absence of diplopia, a horizontal deviation of ≤ 10 prism diopters (PD) and a vertical deviation of ≤ 5 PD at distance in primary and reading positions. RESULTS: Of the 153 GO patients receiving strabismus surgery, 27 cases (20 males, 7 females) underwent reoperation for strabismus, with a reoperation rate of 17.6%. Success rates of reoperation in patients with a previous undercorrection and overcorrection were 45% and 71.4%, respectively. Success rates of rectus recession, rectus resection and anterior advancement were 47.1%, 66.7% and 50%, respectively. Two patients underwent the third surgery. The overall success rate was 51.9%. CONCLUSIONS: Rectus recession is an effective method for GO patients with newly-developed strabismus. Rectus resection may benefit some patients with undercorrection who underwent a maximal degree of rectus recession. Anterior advancement of a previously recessed rectus is effective for cases with overcorrection.

Prolonged hypotony maculopathy following uneventful strabismus surgery.

Hypotony is a rare postoperative complication of strabismus surgery. Resolution has been reported to occur within 1 month of surgery. Here, we describe the case of a 14-year-old boy with prolonged hypotony maculopathy following uneventful bilateral medial rectus recession. The hypotony resolved without long-term sequela after 7 months of treatment with topical steroids and atropine. Ultrasound biomicroscopy revealed a ciliary body effusion, which we hypothesize was the cause of decreased aqueous humor production and hypotony.

[Pediatric myasthenia with ocular involvement]. / Myasthénie chez l'enfant avec atteinte oculaire.

PURPOSE: Myasthenia is a rare disease in children, with an estimated incidence of 1 to 5 per million children. However, the potential severity of its consequences and the existence of specific treatments require prompt diagnosis by pediatric ophthalmologists. METHODS: Retrospective review of patient records. Patients were identified from a rare disease database. Patients under the age of 18 years with confirmed diagnosis of myasthenia and ocular symptoms seen in a specialized clinic between 2005 and 2021 were included. RESULTS: Twenty-six (16 girls) with confirmed myasthenia and ocular symptoms were included. Ten patients had definite autoimmune myasthenia gravis (AIMG); 6 had suspected AIMG with negative antibody testing. Six patients had definite congenital myasthenic syndrome (CMS); 4 had suspected CMS with no evidence of mutation. Mean age at diagnosis of myasthenia was 5 years-3 years and 5 months for CMS and 6 years and 3 months for AIMG. Male to female (M:F) ratio was 6/10 for autoimmune myasthenia gravis and 4/6 for CMS. Ptosis was present in all cases; strabismus in 21 patients (68%). The clinical forms of myasthenia were ocular myasthenia in 12 patients (10 AIMG and 2 CMS), generalized in 12 patients (7 CMS and 5 AIMG) and secondary generalization of ocular myasthenia in 2 patients (2 AIMG). DISCUSSION: These results are based on only 26 cases, which can be explained by the rarity of this diagnosis in children. As in adults, the first signs are often ophthalmologic - ptosis alone or associated with strabismus. Diagnosis is difficult because of the absence of clinical signs, laboratory tests or electrophysiological signs with high sensitivity. Thus, the work-up may remain completely negative in secondarily proven forms. In addition, electroneuromyograms and oculomotor recordings in small children are more difficult to perform than in adults. For these reasons, the clinical examination is essential. In the case of strong suspicion, all additional medical examinations are carried out in a day unit, in order to reach a positive diagnosis of myasthenia. The so-called "congenital" forms, which are genetic, are proportionately higher than in adults, and diagnosis and treatment are often more difficult than in the classic autoimmune forms. CONCLUSION: Myasthenia can affect children from a very young age and can present as ptosis, initially isolated or associated with strabismus. Diagnosis and treatment may be difficult and should be organized in specialized centers.

Characteristics and type of strabismus associated to macular diplopia. Treatment outcomes.

OBJECTIVE: To study the clinical characteristics of macular diplopia, treatment, and outcome. METHODS: Retrospective descriptive study of cases referred to the ocular motility section of a tertiary hospital with diplopia, diagnosed with macular diplopia between 2022-23. The etiology of the macular pathology and the type of associated strabismus were recorded. The result was considered good if the diplopia improved or was eliminated with the medical or surgical treatment. Follow-up time from the onset of diplopia until data collection was recorded. RESULTS: a total of 19 cases comprised the sample (63.2% women), mean age: 67.16 years. Amblyopia (21.1%), high myopia (47.4%), epirretinal membrane (ERM) (36.8%), neovascular membrane (26.3%), macular hole (10.5%), and lamellar (15.8%), and age macular degeneration (5.3%) were registered. The 47.4% had vertical diplopia, horizontal: 5.3 and 47.4% mixed. The mean horizontal deviation was: 7.3 PD (prism diopters) and vertical: 6.22 PD. Ocular extorsion was observed in 26.3%, and intorsion: 5.3%. Torticollis was present in 15.8%. The treatment consisted of strabismus surgery + Botox (15.8%), strabismus surgery (47.4%), medical treatment with Fresnel prims or Scotch cellophane (36.8%). A 68.4% presented a good result at the end of the study. The mean follow-up was 55.58 months. CONCLUSIONS: Misregistration of macular photoreceptors is the most common cause of binocular diplopia in patients with ERM or other macular pathologies. Most complains of vertical or mixed diplopia. Sensorimotor evaluation of these patients should be thorough. Early diagnosis prevents unnecessary prescription of prism glasses. Surgical and/or medical treatment achieves good results in most cases.

Ocular lichen planus as a cause of recurrent restrictive strabismus.

We report the case of an 80-year-old man with restrictive strabismus in lateral gaze following multiple oculoplastic procedures for idiopathic epiphora. Despite excellent initial response to nasal conjunctival recession with lysis of adhesions and a miminal recession of the medial rectus muscle, the patient suffered recurrence of diplopia associated with limitation of abduction due to aggressive, deep, subconjunctival scarring. Given the history of oral lichen planus (LP), the patient was diagnosed with ocular involvement of LP. He underwent a second conjunctival recession, this time accompanied by an intensive LP treatment regimen. Nine months after surgery, he remained diplopia free and orthophoric in primary gaze. Surgeons treating restrictive strabismus in patients with LP should consider implementing systemic and topical immunosuppressive treatment simultaneously with surgical management.

A Case of Superior Oblique Palsy without Brown Syndrome Induced by a Dog Bite.

BACKGROUND Pseudo-Brown syndrome is characterized by dysfunction of the superior oblique tendon-trochlear complex. Canine tooth syndrome, which involves superior oblique palsy with pseudo-Brown syndrome, results from damage to the trochlear and superior oblique tendon from dog bites around the eye. This report describes a variant of canine tooth syndrome without pseudo-Brown syndrome following a dog bite around the left upper eyelid. In this case, magnetic resonance imaging (MRI) facilitated early diagnosis and therapeutic intervention. CASE REPORT A 19-year-old man presented with torsional diplopia following a dog bite around the left upper eyelid and forehead. Five days after the injury, an alternate prism cover test revealed 6 prism diopters (Δ) exotropia and 5Δ left hypertropia. Ocular motility showed no significant limitation in elevation or depression during adduction. MRI performed on the same day showed a high-signal area extending from the superior oblique tendon to the trochlear region and the superior oblique muscle belly of the left eye. A diagnosis of canine tooth syndrome without pseudo-Brown syndrome was made and oral steroids were administered. Ocular alignment did not improve, so left inferior oblique myotomy was performed 7 months after the injury. The patient's cyclovertical diplopia resolved postoperatively. CONCLUSIONS Dog bites around the eye can result in abnormalities of the extraocular muscles. Early MRI may be useful for diagnosis and determining treatment strategies. This report has highlighted the importance of rapid assessment and management of patients with dog bites involving the eye.

Multispecialty Management of Metastatic Colon Adenocarcinoma Involving the Extraocular Muscles: Primary Excision and Simultaneous Treatment of Strabismus With a Review of the Literature.

Metastatic colon adenocarcinoma involving the extraocular muscles is extremely rare. It usually develops following the diagnosis of the systemic disease and therefore, management and treatment require a multispecialty approach. Within this manuscript, we provide a summary of cases of orbital metastasis secondary to colon cancer. We further discuss a detailed case of a 42-year-old male patient who developed recent-onset diplopia in the left gaze. Orbital CT imaging showed a localized, well-circumscribed enlargement of the right medial rectus muscle. The biopsy of the right medial rectus showed adenocarcinoma originating from the gastrointestinal system. Further workup revealed colon adenocarcinoma with multiple metastatic sites. The patient started systemic chemotherapy. After 2 months of chemotherapy (5-fluouracil, oxaliplatin, irinotecan, and leucovorin), all systemic metastatic sites regressed; however, his medial rectus muscle continued to grow, causing compressive optic neuropathy. The patient underwent excisional biopsy of the right medial rectus muscle with simultaneous repair of the strabismus with transposition of superior and inferior recti muscles. He continued with systemic chemotherapy. Follow up in 1 year revealed no local orbital tumor recurrence with excellent visual acuity and no diplopia in primary gaze.

Effect of vertical central plication on vertical deviations.

PURPOSE: To evaluate the surgical effect of the Wright central plication on vertical rectus muscles to correct vertical strabismus. METHODS: In this multicenter, retrospective, observational outcomes study, data were collected from two surgeons in different practice settings (2017-22). All patients who underwent vertical rectus central plication were included; those undergoing any concurrent strabismus surgery for vertical strabismus were excluded. Primary outcome was amount of strabismus correction in prism diopters per vertical rectus central plication. Secondary outcome was to determine factors associated with better or worse surgical outcomes and patient and patient responses. Data were analyzed using descriptive and bivariate statistics. RESULTS: A total of 36 patients were included. Mean age was 60 years. Mean follow-up was 8.4 months. Of the 36 patients, 11 (31%) had idiopathic strabismus, and 7 (19%) had congenital superior oblique palsy. The remainder had a history that included prior ocular surgery, trauma, and Brown syndrome; 16 (44 %) had prior strabismus surgery. Of 31 patients with preoperative diplopia, 23 (74%) had postoperative resolution of diplopia, and 10 of 16 patients with preoperative prisms (63%) no longer required prisms postoperatively. Mean vertical deviation change was 4.7Δ. Subgroup analysis removing patients with congenital superior oblique palsy showed a larger response of 5.5Δ. 78% of patients had a final deviation <5Δ. No complications or induced postoperative diplopia was reported. CONCLUSIONS: In our study cohort, vertical rectus central plication corrected approximately 5Δ (range, 4.5Δ-5.5Δ) of vertical strabismus due to a variety of causes.

Incidence of Strabismus Post-Plaque Brachytherapy in Patients With Uveal Melanoma.

PURPOSE: To determine the incidence and type of strabismus in patients with uveal melanoma treated with plaque brachytherapy. DESIGN: Multicenter, retrospective incidence estimation study. METHODS: A total of 438 eyes of 438 patients with uveal melanoma treated with plaque brachytherapy between October 2011 and May 2021. Intervention was Iodine 125, and Palladium 103 plaque brachytherapy. The variables reviewed included incidence of nonresolving strabismus post-plaque brachytherapy, type of strabismus developed, extraocular muscles operated, and modality of treatment received. RESULTS: A total of 438 patients underwent plaque brachytherapy treatment for uveal melanoma. Eleven patients developed strabismus post-plaque brachytherapy (2.5%, n = 11/438). Of these patients, 5 (1.1%, n = 5/438) developed strabismus immediately postoperation. Specifically, 2 patients (0.5%, n = 2/438) developed strabismus immediately postoperation due to slipped muscles, 2 patients (0.5%, n = 2/438) due to decompensated phorias, and 1 patient (0.5%, n = 1/438) due to a fibrotic muscle. Six patients (1.4%, n = 6/438) developed late-onset sensory strabismus. A total of 355 patients (81.1%, n = 355/438) had their extraocular muscles disinserted during surgery, with the lateral rectus being the most common, accounting for 45.4% (n = 161/355), followed by the superior rectus at 26.8% (n = 95/355). Strabismus surgery was the most common treatment modality, comprising 72.7% (n = 8/11) of patients. CONCLUSIONS: The incidence of strabismus after plaque brachytherapy treatment for uveal melanoma was low and primarily classified as late-onset sensory strabismus. Previous studies may underestimate the long-term incidence of strabismus after plaque brachytherapy by focusing primarily on strabismus present immediately postoperatively.

Ophthalmic sequelae of prematurity in late childhood and adulthood: A review.

Advances in the care of premature infants have resulted in unprecedented rates of survival of these infants into adulthood, including those born at very low gestational ages. Ophthalmologists have historically followed premature infants to assess for the presence of and potential need for treatment of retinopathy of prematurity. However, a growing body of literature suggests that the ophthalmic consequences of prematurity extended beyond retinopathy of prematurity and that ophthalmic sequelae of prematurity can endure through adulthood even among formerly preterm adults who were never diagnosed with retinopathy of prematurity. These abnormalities can include a range of both anterior segment and posterior segment sequelae, including higher rates of corneal aberrations, ocular hypertension, strabismus, foveal anomalies, and retinal tears and detachments. This review aims to summarise this literature, underscoring the importance of lifelong examinations and regular monitoring for these complications among adults who were born prematurely.

Pediatric myopic strabismus fixus: clinical features and surgical outcomes of silicone band loop myopexy.

PURPOSE: To evaluate the clinical profile of myopic strabismus fixus (MSF) in children and surgical outcomes of silicone band loop myopexy. METHOD: We retrospectively reviewed records of children presenting with MSF who underwent silicone band loop myopexy between January 2008 and December 2020 at a tertiary eye care center. Data concerning demographics, refractive error, axial length, extra-ocular motility, and ocular alignment pre-operatively and post-operatively, intra- and post-operative complications, ocular and systemic associations, were evaluated. The long-term effects of band loop myopexy on ocular alignment stability, motility improvement, and myopia progression were analyzed. Surgical outcome was defined as post-operative orthotropia or heterotropia less than or equal to 20 PD. RESULTS: A total of0 eyes of 7 patients (median age: 5 years; 5 boys and 2 girls) who underwent band loop myopexy were included in the study. Among them, three children underwent bilateral and four children underwent unilateral band loop myopexy. Medial rectus recession was performed only in two patients as a part of initial procedure. The median follow-up duration was 7 years. Most of the children, i.e. six of them presented with esotropia-hypotropia and only one patient presented with exotropia-hypotropia complex. The median pre-operative measurements were esotropia of 62.5 PD, hypotropia of5 PD, and exotropia of4 PD. Postoperative average primary position deviation measured was close to 9-10 PD of esotropia. The overall motility improved to -1 from -3. CONCLUSION: The clinical profile of MSF in children is almost similar to adults. This condition is a rare entity among adults as well as children. Majority of children with MSF presented with esotropia-hypotropia complex. Silicone band loop myopexy with or without medial rectus recession proves to be a reliable surgical procedure as it provides stable outcomes in terms of ocular alignment and motility among children.

The influence of orbital architecture on strabismus in craniosynostosis.

PURPOSE: To better characterize the correlation of bony orbital dysmorphology with strabismus in craniosynostosis. METHODS: The medical records of patients with craniosynostosis with and without strabismus seen at Rady Children's Hospital (San Diego, CA) from March 2020 to January 2022 were reviewed retrospectively in this masked, case-control study. Computed tomography scans of the orbits were analyzed to obtain dimensions of the orbital entrance and orbital cone. Primary outcome was correlation of strabismus with orbital measurements. RESULTS: A total of 30 orbits from 15 patients with strabismus and 15 controls were included. Craniofacial disorders included in the study were nonsyndromic craniosynostosis (63%), Crouzon syndrome (13%), Apert syndrome (13%), and Pfeiffer syndrome (10%). Orbital index (height:width ratio) (P = 0.01) and medial orbital wall angle (P = 0.04) were found to differ significantly between the strabismus and control groups. CONCLUSIONS: In our small cohort, bony orbital dimensions, including the ratio of orbital height to width and bowing of the medial orbital wall, were associated with strabismus in craniosynostosis.

An unexpected turn: a case of torsional anomalous retinal correspondence after myectomy of previously anteriorized inferior oblique muscles in a child.

We report a case of torsional diplopia caused by presumed torsional anomalous retinal correspondence after myectomy of previously asymmetrically anteriorized inferior oblique muscles for inferior oblique overaction. Given this patient's experience, it may be prudent to operate with caution on previously anteriorized inferior oblique muscles, especially when anteriorization is performed at a very young age.

"Strabismus in children" is not simple - bilateral persistent hyperplasia primary vitreous: a case report.

Persistent hyperplastic primary vitreous (PHPV) is a rare congenital developmental ocular disorder caused by incomplete regression of the embryonic hyaloid vasculature; bilateral presentations are even rarer. We report a 6-year-old child with bilateral PHPV who visited our hospital for strabismus, without exhibiting leukocoria, microphthalmia, and systemic diseases. These unique characteristics distinguish our case from other cases of PHPV. It is crucial to increase awareness of congenital eye disease in children and the importance of performing fundus examination with the pupils dilated.

The effect of previous orbital decompression on outcomes of rectus muscle recession surgery in patients with thyroid ophthalmopathy.

PURPOSE: To evaluate the effect of previous orbital decompression on outcomes of rectus muscle recession surgery in patients with thyroid-associated ophthalmopathy. METHODS: This retrospective study enrolled 55 eyes of 33 patients treated in our hospital for restrictive strabismus caused by thyroid-associated ophthalmopathy. We performed muscle recession for the obviously restricted extraocular muscles, with 6 weeks of follow-up. Surgical outcomes were compared between the orbital decompression group (DG, n=15) and non-orbital decompression group (NDG, n=18). RESULTS: A total of 33 patients with Graves' ophthalmopathy who underwent rectus muscle recession surgery were included. Of these, 15 patients had undergone orbital decompression prior to strabismus surgery, and 18 had not. The two groups did not differ in terms of the preoperative horizontal or vertical ocular deviation, degree of restriction of eye movement, degree of diplopia, or mean number of muscles that underwent surgery (P>0.05). There was no significant difference in the preoperative horizontal or vertical ocular deviation, level of eye movement restriction, degree of diplopia and the success rate of the surgery (P>0.05). CONCLUSION: Rectus muscle recession surgery in patients with thyroid-associated ophthalmopathy during the quiescent period could improve the ocular deviation and diplopia, and orbital decompression performed before strabismus surgery had no significant effect on surgical technique or outcomes of rectus muscle recession surgery.

A patient with congenitally restrictive strabismus and contralateral secondary muscle enlargement.

AIM: To report the case of a congenital restrictive strabismus with a contralateral enlargement of extraocular muscles. CASE: The clinical presentation, findings, and postoperative outcomes of a 4 years old boy referred to evaluation for presenting a left eye deviation since birth are detailed. CONCLUSION: A unilateral congenital restrictive strabismus (congenital hypotropia and esotropia) can be the cause of contralateral secondary enlarged extraocular muscle and It must be included in the differential diagnosis.