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Therapeutic Advances in Ophthalmology 2024The current data on various aspects of Brown syndrome are limited and sporadic. This review provides a coherent and comprehensive review of basic features, etiology,... (Review)
Review
The current data on various aspects of Brown syndrome are limited and sporadic. This review provides a coherent and comprehensive review of basic features, etiology, classification, differential diagnosis, and different management strategies of patients with Brown syndrome. In this topical review, PubMed, Scopus, and Google Scholar search engines were searched for papers, published between 1950 and January 2023 based on the keywords of this article. The related articles were collected, summarized, categorized, assessed, concluded, and presented. Brown syndrome is identified by restricted passive and active elevation of the eye in adduction. The condition is divided into congenital and acquired causes. The clinical features result from a restricted motion of the superior oblique tendon sheath through the trochlea while trying to look up in adduction. The newest explanation of the underlying pathophysiology has been explained as the presence of a fibrotic strand in the superior oblique muscle tendon with variable insertion sites which creates various elevation deficits seen in Brown syndrome. The most common clinical features include the presence of an abnormal head posture, V-pattern strabismus, and hypotropia in the primary position. Management of Brown syndrome includes watchful observation, surgical, and non-surgical procedures. Some cases might resolve spontaneously without any intervention; however, some acquired cases might require systemic and/or intra-trochlear steroid administration to treat the underlying causes. Surgical procedures such as superior oblique tenectomy and using a silicon tendon expander are indicated in the presence of hypotropia and significant abnormal head posture in the primary position.
PubMed: 38406627
DOI: 10.1177/25158414231222118 -
Acta Ophthalmologica Mar 2021To elucidate the patterns of strabismus and ophthalmoplegia associated with chronic progressive external ophthalmoplegia (CPEO) confirmed by mitochondrial DNA (mtDNA)...
PURPOSE
To elucidate the patterns of strabismus and ophthalmoplegia associated with chronic progressive external ophthalmoplegia (CPEO) confirmed by mitochondrial DNA (mtDNA) deletions in Asians.
METHODS
A total of 10 patients confirmed to have mtDNA deletion associated with CPEO were included. Long-range PCR encompassing the entire mtDNA was carried out. In the cases with mtDNA deletion, the exact deletion ranges of mtDNA were identified by sequencing. A full ophthalmologic examination including prism and alternate cover test in the primary position, evaluation of ductions and versions, and binocularity was performed in 10 patients with confirmed mtDNA deletions associated with CPEO.
RESULTS
All of the patients showed ophthalmoplegia as well as ptosis, even after eyelid surgeries. Ophthalmoplegia was symmetric between both eyes in nine patients (90%) while one patient (10%) showed asymmetric ophthalmoplegia with esotropia and left hypotropia. Among the nine patients with symmetric involvement, four patients (44%) showed exotropia, three (33%) had exotropia with vertical deviation, and the remaining two patients (22%) showed orthotropia. Five out of 10 patients (50%) complained of diplopia associated with strabismus, four of whom (80%) had vertical deviation. Three out of five patients (60%) without diplopia showed exotropia of 20 prism diopters (PD) to 50 PD.
CONCLUSIONS
Exotropia with/without vertical deviation is the most common form of strabismus in Asian patients with CPEO and only one of them showed a small angle of esotropia. Ophthalmoplegia could be asymmetric in 10% of CPEO patients.
Topics: Adolescent; Adult; Child; Chronic Disease; DNA, Mitochondrial; Disease Progression; Female; Gene Deletion; Humans; Male; Middle Aged; Ophthalmoplegia; Retrospective Studies; Strabismus; Young Adult
PubMed: 33191655
DOI: 10.1111/aos.14558 -
The Cochrane Database of Systematic... Nov 2015The term "strabismus" describes misalignment of the eyes. One or both eyes may deviate inward, outward, upward, or downward. Dissociated vertical deviation (DVD) is a... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
The term "strabismus" describes misalignment of the eyes. One or both eyes may deviate inward, outward, upward, or downward. Dissociated vertical deviation (DVD) is a well-recognized type of upward drifting of one or both eyes, which can occur in children or adults. DVD often develops in the context of infantile- or childhood-onset horizontal strabismus, either esotropia (inward-turning) or exotropia (outward-turning). For some individuals, DVD remains controlled and can only be detected during clinical testing. For others, DVD becomes spontaneously "manifest" and the eye drifts up of its own accord. Spontaneously manifest DVD can be difficult to control and often causes psychosocial concerns. Traditionally, DVD has been thought to be asymptomatic, although some individuals have double vision. More recently it has been suggested that individuals with DVD may also suffer from eyestrain. Treatment for DVD may be sought either due to psychosocial concerns or because of these symptoms. The standard treatment for DVD is a surgical procedure; non-surgical treatments are offered less commonly. Although there are many studies evaluating different management options for the correction of DVD, a lack of clarity remains regarding which treatments are most effective.
OBJECTIVES
The objective of this review was to determine the effectiveness and safety of various surgical and non-surgical interventions in randomized controlled trials of participants with DVD.
SEARCH METHODS
We searched CENTRAL (which contains the Cochrane Eyes and Vision Trials Register) (2015, Issue 8), Ovid MEDLINE, Ovid MEDLINE In-Process and Other Non-Indexed Citations, Ovid MEDLINE Daily, Ovid OLDMEDLINE (January 1946 to August 2015), EMBASE (January 1980 to August 2015), PubMed (1948 to August 2015), Latin American and Caribbean Health Sciences Literature Database (LILACS) (1982 to August 2015), the metaRegister of Controlled Trials (mRCT) (www.controlled-trials.com) (last searched 3 February 2014), ClinicalTrials.gov (www.clinicaltrials.gov), and the WHO International Clinical Trials Registry Platform (ICTRP) (www.who.int/ictrp/search/en). We did not use any date or language restrictions in the electronic searches for trials. We last searched the electronic databases on 3 August 2015.
SELECTION CRITERIA
We included randomized controlled trials (RCTs) of surgical and non-surgical interventions for the correction of DVD.
DATA COLLECTION AND ANALYSIS
We used standard procedures expected by Cochrane. Two review authors independently completed eligibility screening, data abstraction, 'Risk of bias' assessment, and grading of the evidence.
MAIN RESULTS
We found four RCTs eligible for inclusion in this review (248 eyes of 151 participants between the ages of 6 months to 22 years). All trials were assessed as having unclear risk of bias overall due to insufficient reporting of study methods. One trial was conducted in Canada and compared anteriorization of the inferior oblique muscle with resection versus anteriorization of the inferior oblique muscle alone; one in the USA compared superior rectus recession with posterior fixation suture versus superior rectus recession alone; and two in the Czech Republic compared anteriorization of the inferior oblique muscle versus myectomy of the inferior oblique muscle.Only one trial reported data that allowed analysis of the primary outcome for this review, the proportion of participants with treatment success. The difference between inferior oblique anteriorization plus resection versus inferior oblique anteriorization alone was uncertain when measured at least four months postoperatively (risk ratio 1.13, 95% confidence interval 0.60 to 2.11, 30 participants, very low-quality evidence). Three trials measured the magnitude of hyperdeviation, but did not provide sufficient data for analysis. All four trials reported a relatively low rate of adverse events; hypotropia, limited elevation, and need for repeat surgery were reported as adverse events associated with some of the surgical interventions. No trials reported any other secondary outcome specified for our review.
AUTHORS' CONCLUSIONS
The four trials included in this review assessed the effectiveness of five different surgical procedures for the treatment of DVD. Nevertheless, insufficient reporting of study methods and data led to methodological concerns that undermine the conclusions of all studies. There is a pressing need for carefully executed RCTs of treatment for DVD in order to improve the evidence for the optimal management of this condition.
Topics: Adolescent; Child; Child, Preschool; Humans; Infant; Oculomotor Muscles; Randomized Controlled Trials as Topic; Strabismus; Treatment Outcome; Young Adult
PubMed: 26587695
DOI: 10.1002/14651858.CD010868.pub2 -
Beyoglu Eye Journal 2021The goal of this study was to evaluate surgical techniques and outcomes in patients with Brown's syndrome.
OBJECTIVES
The goal of this study was to evaluate surgical techniques and outcomes in patients with Brown's syndrome.
METHODS
A retrospective review was conducted of patients who underwent surgery of the superior oblique (SO) muscle between 2003 and 2011 at a referral center.
RESULTS
In all, 190 patients (111 female and 79 male) with an age range of 4-50 years were included in the study. The right eye was affected in 98 patients, and the left eye in 92 patients. Abnormal head posture (AHP), ocular movement (OM), and hypotropia were assessed. The greatest improvement of AHP was seen following an SO temporal tenotomy (91%). Patients with a -4 limitation achieved full OM after a SO temporal tenotomy.
CONCLUSION
Temporal tenotomy provided the best improvement in limitation of elevation in adduction.
PubMed: 35005520
DOI: 10.14744/bej.2021.35693 -
European Journal of Ophthalmology Jul 2020To assess the efficacy of "Yokoyama Procedure," on non-highly myopic patients with acquired esotropia and hypotropia.
OBJECTIVE
To assess the efficacy of "Yokoyama Procedure," on non-highly myopic patients with acquired esotropia and hypotropia.
METHODS
The study involved 10 eyes of 5 patients with eso-hypotropia. Inclusion criteria were acquired esotropic-hypotropic strabismus with lateral rectus inferior displacement and superior rectus nasal displacement confirmed by magnetic resonance imaging, refractive errors between ±6 D, and axial length < 27 mm. Range of full duction movements and maximum angles of abduction-sursumduction was measured in each eye before and after surgery. All patients underwent T1- and T2-weighted magnetic resonance imaging. The surgery was aimed at creating a junction between the muscle bellies of the superior and lateral rectus muscles. This junction was made approximately 14 mm behind the insertions using a non-absorbable mersilene 5/0 suture (Yokoyama procedure).
RESULTS
Mean patient age was 64.8 ± 4.8 years. The mean globe axial length was 25.4 ± 0.76 mm and a mean corresponding spherical equivalent refraction of -3.7 ± 1.7 D was observed. Eight eyes on 10 had mild limitation in abduction, while the remaining 2 had no limitation. Three out of 10 eyes showed a moderate limited sursumduction, 5 eyes were categorized as mild, and the remaining 2 had no limitation. No evident post-operative limitation was present in any eye, in both abduction and sursumduction (p < 0.01). Pre-operative esotropia and hypotropia were, respectively, 32 ± 11 prismatic diopters and 25 ± 5 prismatic diopters, and they were significantly reduced after surgery as 9 ± 1.7 prismatic diopters and 6 ±1 prismatic diopters (p = 0.043), respectively.
CONCLUSION
Yokoyama procedure is an effective, fast, reversible procedure to face eso-hypotropic acquired strabismus, even in patients with a clear magnetic resonance imaging displacement of superior and lateral rectus muscles, and absence of globe dislocation and of elevated myopia.
Topics: Aged; Esotropia; Female; Follow-Up Studies; Humans; Magnetic Resonance Imaging; Male; Middle Aged; Myopia; Oculomotor Muscles; Ophthalmologic Surgical Procedures; Strabismus; Treatment Outcome; Vision, Binocular; Visual Acuity
PubMed: 31106643
DOI: 10.1177/1120672119850684 -
Neuro-ophthalmology (Aeolus Press) Oct 2015A 14-year-old boy presented with left ptosis and squint since his childhood. In primary position he had left hypotropia with ptosis (levator function 2 mm). With...
A 14-year-old boy presented with left ptosis and squint since his childhood. In primary position he had left hypotropia with ptosis (levator function 2 mm). With elevation of the right eye, the left eye depresses and adducts; with depression of the right eye, the left eye elevates and abducts. Forced duction test revealed no restrictive pattern. The synergistic divergence described in the literature was unidirectional and wholly compatible with inferior rectus co-contraction. But this case is bidirectional, and the aberrant upward movement cannot be explained by rectus muscle co-contraction. The two vertical rectus muscles show reciprocal misinnervation, or this effect could be coming from co-contraction of one of the oblique muscles for the movement in one direction.
PubMed: 27928360
DOI: 10.3109/01658107.2015.1056379 -
Indian Journal of Ophthalmology Mar 2023To assess the clinical profiles and outcomes of strabismus in pediatric patients with orbital wall fractures.
PURPOSE
To assess the clinical profiles and outcomes of strabismus in pediatric patients with orbital wall fractures.
METHODS
A retrospective interventional study of all consecutive children of age ≤16 years who presented with traumatic orbital wall fractures with and without resultant strabismus was conducted. The details of patient demographics, clinical features, interventions, and outcomes were obtained.
RESULTS
Forty-three children presented with traumatic orbital fractures to a tertiary care center. The mean age at presentation was 11 years and there was a male predominance (72.09%). Isolated floor fracture involvement was the most common (n = 24, 55.81%), and almost half of the children had a white-eyed or trapdoor fracture (n = 21, 48.83%). Twenty-six (60.46%) children had surgical repair of fracture(s). Manifest strabismus following orbital fracture was documented in 12 children (27.90%). Of these, an exotropia was noted in seven (58.33%), hypotropia in two (16.67%), hypertropia in one (8.33%), and esotropia in one patient (8.33%), while an exotropia with hypotropia was noted in one patient (8.33%). Restrictive nature of strabismus due to either muscle entrapment or local trauma was more commonly observed in 11/12 patients (91.66%). Primary position diplopia before orbital wall fracture repair was observed in four children and after repair was observed in two children with manifest strabismus. Four children underwent strabismus surgery post-fracture repair.
CONCLUSION
An improvement in strabismus and ocular motility was noted in majority of the patients after fracture repair. The few that underwent strabismus surgery had a restrictive nature of strabismus. Trapdoor fractures and the nature of trauma in children vary compared to adults. Persistence of strabismus may be due to an excessive time interval between trauma and fracture repair or the extensive nature of trauma.
Topics: Adult; Humans; Child; Male; Adolescent; Female; Orbital Fractures; Retrospective Studies; Strabismus; Esotropia; Exotropia
PubMed: 36872720
DOI: 10.4103/ijo.IJO_1929_22 -
American Journal of Ophthalmology Case... Jun 2018We present the first reported case of Waldenstrom macroglobulinemia in the right superior rectus causing diplopia.
PURPOSE
We present the first reported case of Waldenstrom macroglobulinemia in the right superior rectus causing diplopia.
OBSERVATIONS
A 72-year-old man with a 6-month history of untreated asymptomatic Waldenstrom macroglobulinemia presented with 2 years of diagonal binocular diplopia that was previously thought to be due to ocular myasthenia gravis. Examination showed mild right proptosis and right hypotropia, and MRI revealed a focal lesion of the right superior rectus muscle. Orbital biopsy was performed, and histopathology showed lymphoplasmacytic infiltration among the skeletal muscle fibers of the rectus muscle. Immunostaining confirmed a B-cell preponderance, along with more extensive staining for IgM than IgG, and in situ hybridization confirmed lambda restriction. These findings corresponded with those of his previous bone marrow biopsy, confirming Waldenstrom macroglobulinemia as the etiology for the extraocular muscle mass.
CONCLUSIONS AND IMPORTANCE
Lymphoma of an extraocular muscle is a rare manifestation of orbital lymphoma, and the tumors are usually mucosa-associated lymphoid tissue (MALT) lymphomas (i.e. extranodal marginal zone lymphomas). There are 4 previous reports of lymphoplasmacytic lymphoma of an extraocular muscle; however this is the first reported case of such a lesion in a patient with concurrent Waldenstrom macroglobulinemia at the time of diagnosis.
PubMed: 29780960
DOI: 10.1016/j.ajoc.2018.04.008 -
Case Reports in Ophthalmology 2022The aim of this paper was to report the case of a patient with orbital inflammation and dacryoadenitis after COVID-19 vaccination. During the COVID-19 pandemic, we...
The aim of this paper was to report the case of a patient with orbital inflammation and dacryoadenitis after COVID-19 vaccination. During the COVID-19 pandemic, we noticed an increase of post-viral syndromes, both linked to infection and to vaccination. A 53-year-old male presented with proptosis, chemosis, hypotropia, and ophthalmoplegia of the right eye, 1 day after his COVID-19 booster shot. Anecdotally, he suffered similar symptoms after his initial two vaccinations. Idiopathic orbital inflammation and dacryoadenitis were diagnosed, and the patient was successfully treated with oral steroids. Orbital inflammation and dacryoadenitis after infection or vaccination are no new entities, but due to the scale of the current pandemic and the associated vaccination programs, these rare ocular diseases could be encountered more often.
PubMed: 36845447
DOI: 10.1159/000526225 -
Arquivos Brasileiros de Oftalmologia 2021This study was conducted to further define the specific clinical characteristics of patients with Brown syndrome and evaluate the outcomes of superior oblique tenotomy...
PURPOSE
This study was conducted to further define the specific clinical characteristics of patients with Brown syndrome and evaluate the outcomes of superior oblique tenotomy in its surgical management.
METHODS
A retrospective analysis of the medical charts of 45 patients with Brown syndrome was performed, which revealed that 11 patients underwent superior oblique tenotomy due to abnormal head posture and/or hypotropia and 1 patient underwent bilateral superior oblique tendon elongation with a silicone band due to abnormal head posture. In the last patient, silicone bands were removed at the postoperative 3rd month due to the lack of improvement in the abnormal head posture and the limitation of elevation in adduction. Simultaneous horizontal rectus muscle surgery was performed in four patients.
RESULTS
There was a predominance of female gender, right eye, congenital form, unilaterality, A-pattern, and an abnormal head posture type with a combination of chin up and head tilting. Bilateral form was observed only in female patients. Amblyopia was detected in two patients. Among patients aged >5 years, 40% had reduced stereopsis. Abnormal head posture was found in 60% of patients. More than half of them were diagnosed with a vertical and/or horizontal deviation. Tenotomy procedure eliminated the abnormal head posture in all patients and significantly improved the mean limitation of elevation in adduction and hypotropia (p=0.001, p=0.012). Two patients developed inferior oblique overaction in the operated eye. There was complete spontaneous resolution in two patients.
CONCLUSIONS
The clinical features of patients with Brown syndrome in our study are considerably consistent with those of previous reports. The present study demonstrated the effectiveness of superior oblique tenotomy with less overcorrection in the surgical treatment of Brown syndrome.
Topics: Aged; Female; Humans; Ocular Motility Disorders; Oculomotor Muscles; Ophthalmologic Surgical Procedures; Retrospective Studies; Strabismus; Tenotomy
PubMed: 33567010
DOI: 10.5935/0004-2749.20210021