Brown Syndrome Research Articles

Full-genome sequencing of German rabbit haemorrhagic disease virus uncovers recombination between RHDV (GI.2) and EBHSV (GII.1).

Rabbit haemorrhagic disease virus (RHDV; genotypes GI.1 and GI.2) and European brown hare syndrome virus (EBHSV; genotype GII.1) are caliciviruses belonging to the genus Lagovirus. These viruses pose a serious threat to wild and domestic rabbit and hare populations around the world. In recent years, an expanding genetic diversity has been described within the genus, with recombination events occurring between the different genotypes. Here, we generated and analysed 56 full-genome sequences of RHDV and EBHSV from rabbit and hare livers, collected in Germany between the years 2013 and 2020. We could show that genotype Gl.2 (RHDV-2) almost entirely replaced Gl.1 (classical RHDV) in the German rabbit population. However, GI.1 is still present in Germany and has to be included into disease control and vaccination strategies. Three recombinant strains were identified from rabbit samples that contain the structural genes of genotype Gl.2 and the non-structural genes of genotype Gl.1b. Of special interest is the finding that sequences from two hare samples showed recombination events between structural genes of RHDV Gl.2 and non-structural genes of EBHSV GII.1, a recombination between different genogroups that has not been described before. These findings lead to the assumption that also a recombination of the non-structural genes of RHDV Gl.2 with the structural genes of EBHSV Gll.1 might be possible and therefore increase the potential genetic variability of lagoviruses immensely. Our findings underline the importance of whole genome analysis with next-generation sequencing technology as one of new tools now available for in-depth studies that allow in depth molecular epidemiology with continuous monitoring of the genetic variability of viruses that would otherwise likely stay undetected if only routine diagnostic assays are used.

Brown Syndrome from Local Anesthesia for Inferior Orbital Fat Decompression

Patient: Female, 36-year-oldFinal Diagnosis: Acquired Brown syndromeSymptoms: Diplopia • vertical diplopiaMedication: —Clinical Procedure: Local anesthesia • orbital fat decompressionSpecialty: OphthalmologyObjective:Unusual or unexpected effect of treatmentBackground:Vertical diplopia that follows local anesthesia is usually due to inferior rectus muscle fibrosis. Here, we report a rare case of acquired Brown syndrome following local anesthesia.Case Report:A 36-year-old woman underwent right inferior orbital fat decompression under local anesthesia. On the first postoperative day, she developed vertical diplopia. She had left hypertropia, which increased on left gaze, with limitation of elevation of the right eye on attempted adduction. Forced duction test of the right eye revealed resistance on elevation in adduction. Magnetic resonance imaging showed signal alteration, thickening, and irregularity involving the right superior oblique tendon and trochlea region. The diagnosis of iatrogenic Brown syndrome was made. Then, a single dose of 10 mg triamcinolone injection was given near the intratrochlear region. On follow-up, complete resolution of diplopia on primary gaze occurred 12 weeks after the incident.Conclusions:The reported case highlights that local anesthesia carries a risk of Brown syndrome. We believe bupivacaine-induced superior oblique hypertrophy is the underlying mechanism. The patient showed excellent outcome after medical management, with no surgical intervention required after 3 months of follow-up.

Surgical reposition of the trochlea in a case of acquired Brown's Syndrome

Surgical reposition of the trochlea in a case of acquired Brown's Syndrome

European Brown hare (Lepus europaeus) as a source of emerging and re-emerging pathogens of Public Health importance: A review.

European brown hare (Lepus europaeus, EBH) is probably the most important game animal in Europe throughout its historical distribution. The decline in its populations across its geographic range in Europe have been attributed to factors such as reproductive rate and the ability for adaptation, climate, feed availability, predators, anthropogenic factors and diseases. Apart from common diseases of hares with a high impact on their mortality such as European Brown hare Syndrome, EBH has been involved in the epidemiology of pathogens with zoonotic potential. In this work, the role of EBH as a source of Crimean–Congo haemorrhagic fever virus (CCHFV), Hepatitis E virus (HEV), Yersinia spp., Brucella spp., Francisella tularensis, Toxoplasma gondii and Leishmania infantum is discussed. Hares may significantly contribute to the epidemiology of important emerging zoonotic pathogens through maintenance of high endemicity levels as in the case of CCHFV, as a reservoir of important pathogens such as Yersinia spp., B. suis, F. tularensis and L. infantum and as a potential source of T. gondii for other animals, especially for carnivores but also for humans. However, EBH may also be a host of minor importance as in the case of HEV. The continuous surveillance of hare populations will enable the collection of information on the population health status and the pathogens currently circulating in the area posing risk for wildlife, domestic animals and humans. The possible live animal translocations of infected hares, the fact that this species acts as a host of vectors (fleas, ticks, mosquitoes and sandflies) and the prey of carnivores and omnivores that travel in great distances getting into contact with domestic animals and humans, further highlights the need to be included in surveillance studies. Besides, the hunter‐harvested EBH population is an excellent indicator for recent pathogen transmission due to its short lifespan.

Brown Bowel Syndrome: A Multi-institutional Case Series.

Brown bowel syndrome (BBS) is a rare condition associated with vitamin E deficiency and defined by prominent lipofuscin deposition in the muscularis propria. Eight unique cases of BBS were identified: 5 men and 3 women (mean age=58.6 y). Pertinent comorbidities included bariatric surgery=2, malnourishment=2, Crohn=2, cystic fibrosis=1, alcohol and cocaine abuse=1, and prior small bowel resections=1. Presenting symptoms included abdominal pain=3, bleeding=1, nausea and vomiting=1, and nonresponsiveness=1. Imaging studies were often abnormal: thickened bowel wall=3 (1 with a mass), small bowel obstruction=2, and edematous and dilated bowel wall=2. Most specimens were surgical resections (n=7, autopsy=1): extended right colectomy=2, small bowel only=5 (terminal ileum=3, jejunum=2). Two specimens were grossly described as mahogany, and 1 case contained a perforation. Histologic sections of all cases showed finely granular, brown cytoplasmic pigment in smooth muscle cells on hematoxylin and eosin. This pigment was most conspicuous in the muscularis propria (small bowel>colon), and it was not identified in the mucosa. The pigment was reactive with Fontana-Masson, carbol lipofuscin, Periodic acid-Schiff, and Periodic acid-Schiff with diastase, and electron microscopy was compatible with lipofuscin. The mean clinical follow-up was 208 weeks: 1 patient died of complications of encephalitis, the others were alive and well. BBS is important to recognize because it is linked with malnutrition, specifically vitamin E deficiency, and it can (rarely) clinically simulate malignancy. The diagnosis is based on the identification of the lipofuscin pigment in the cytoplasm of smooth muscle cells, which is most easily seen in the muscularis propria of the small bowel.

The Spectrum of Underlying Diseases in Children with Torticollis.

To investigate the underlying conditions in children with torticollis. Between May 2016 and December 2019, 24 patients (10 girls and 14 boys; mean age, 8 years) presenting with twisted neck, neck pain, weakness of extremities, imbalance, and gait disorder were evaluated retrospectively. Five of the patients had cranial pathologies (cerebellar anaplastic ependymoma and medulloblastoma, brain stem glioma, atypical teratoid rhabdoid tumor, and acute disseminated encephalomyelitis), and five of the patients had spinal pathologies (idiopathic intervertebral disc calcification, vertebral hemangiomatosis, compression fracture, multiple hereditary exostoses, and Langerhans cell histiocytosis at C4). Six of the patients had ocular pathologies (strabismus, Duane syndrome, and Brown syndrome each in two patients). Four patients had otorhinolaryngological infections (Sandifer syndrome, esophageal atresia, reflux, and spasmus nutans, with one patient each). Detailed clinical physical examination and necessary laboratory investigation were performed for all patients. Torticollis is a sign that is not always innocent and may herald an underlying severe disease. Misdiagnosis can lead to wrong and unnecessary surgical procedures and treatments, and sometimes, the results can be damaging due to underlying severe conditions if diagnosed late. In addition, we first report a case of vertebral hemangiomatosis and temporomandibular joint ankylosis that presented with torticollis in the English medical literature.

Brown Sequard's syndrome (spinal hemiplegia) and calcified cervical disc herniation

Brown-Sequard´s syndrome (BSS) is an unusual clinical entity caused by damage to one half of the spinal cord mimicking a classic hemiplegia. This syndrome is characterized by ipslateral loss of motor function (paralysis), proprioception, and vibratory sensation, combined with contralateral loss of pain and temperature sensation. Spinal cord injuries and tumoral diseases were the most frequent etiologies associated with BSS. Spontaneous cervical disc herniation has rarely been considered.

Mysterious Syndrome Causing High Mortality in Wild Brown Trout in Eastern Switzerland, Similar to Proliferative Darkening Syndrome – Pathology and Possible Causes

Introduction: In the Thur, a river situated in the Eastern part of Switzerland, massive mortalities have occurred regularly in brown trout since 2016. Dead fish were recorded in a period of a few weeks in July/August and with a sharp demarcation between affected and unaffected river stretches. The majority of affected animals were 1-year old. Materials and Methods: From June to November 2018, brown and rainbow trout were sampled from the Thur. For each species, five fish were sampled every 2 weeks. Parasitology, bacteriology, virology and histology were performed. The water temperature was measured regularly. Water samples were taken every 10 minutes and pooled for daily samples for a period of 6 months. Non-target screening, target screening and suspect screening were performed. Results: Macroscopically, clinically ill fish showed dark colouration and apathy. No consistent pathogenic agent was isolated. By histology, extensive liver necrosis, haemorrhage and severe lymphohistiocytic myocarditis were diagnosed exclusively in brown trout. The first lesions were found in mid-August, while the highest degree of alterations occurred from the end of August to October. Full recovery did not occur until final sampling at the end of November. As alterations were in agreement with those recently described for brown trout proliferative darkening syndrome (PDS) in Germany, severely affected specimens were analysed for piscine reovirus 1 and 3, agents proposed to be involved in PDS. None of the examined samples were found to be positive. Discussion: Up to now, the cause of the described syndrome remains unclear. Results from water analyses are still outstanding.

Brown's atavistic superior oblique syndrome: etiology of different types of motility disorders in congenital Brown's syndrome

The congenital Brown syndrome is characterized by a mechanical limitation of elevation in adduction, with an orthophoria in down gaze. Brown postulated a shortened superior oblique tendon sheath as the cause of the limitation but this was disproved by Parks et al. in 1975 and the origin of Brown syndrome remains unclear. In recent years, acongenital dysinnervation has been discussed; however, this does not explain the full spectrum of abnormalities and especially contradicts the unlimited depression in adduction seen in Brown syndrome. Surgical exploration in Brown true typical cases reveals a fibrotic strand, typically located at the posterior margin of the superior oblique tendon. This strand originates from the trochlear area and has a common insertion with the superior oblique tendon posterior to the equator into the globe. It may represent an atavistic superior oblique muscle as described by Fink in various animals. They do not have a trochlea but a superior oblique muscle originating in the anterior superior nasal orbit. Afibrotic strand was also surgically revealed in two cases of atypical Brown syndrome. In the first case an elevation deficit-as in Brown true atypical cases-also present in abduction could be explained by an unusual insertion of the fibrotic strand anterior to the equator. The second case showed a fibrotic strand which was completely separated from the superior oblique tendon and inserted far posterior to the equator nasal to the superior rectus muscle. This finding had not been previously described and explained the total elevation restriction which was suddenly in > 30° adduction and the Y‑pattern exotropia which increased in adduction and decreased in abduction. A10 mm excision of the fibrotic strand from the insertion gives the best results from all procedures. The residual limitation of active elevation in adduction improved with gaze exercises mostly after more than 1year. The fibrotic strand, an atavistic superior oblique muscle, not only explains the typical Brown syndrome but also-by its variable insertion-different patterns of elevation deficits seen in atypical Brown syndrome. A10 mm excision of the strand gives good functional results of abnormal head position (immediate in most cases) and even elevation in adduction (after 1year in most cases).

An Interesting Case of Acquired Brown Syndrome - Superior Oblique Myocysticercosis

An Interesting Case of Acquired Brown Syndrome - Superior Oblique Myocysticercosis

Intratrochlear steroid injections in acquired Brown syndrome—a case series

To present our experience in the treatment of children with acquired Brown syndrome by means of intratrochlear injection of betamethasone. The medical records of patients treated with intratrochlear betamethasone in 2016 at the Aravind Eye Hospital, Madurai, were reviewed retrospectively. The following data were collected: pre- and postoperative orthoptic work-up, blood work, and neuroimaging. Betamethasone injection was administered 2-8weeks following onset of symptoms. Five children (4 girls), 1.5-15years of age, were included. During the postoperative period, abnormal head posture and elevation in adduction improved in 4 subjects but did not resolve completely. The median vertical deviation was 11.5Δ preoperatively and reduced to 3.5Δ postoperatively. A significant reduction in deviation was demonstrable on diplopia and Hess charting in 2 of the older children. Subject 2, who did not show improvement after injection, was prescribed prism glasses and became diplopia free. In this case series, children with acquired Brown syndrome of idiopathic or presumed inflammatory etiology showed significant reduction in deviation and symptoms following intratrochlear injection of betamethasone. We recommend that this treatment be considered for children affected by acquired Brown syndrome, especially those in the amblyogenic age group.

The discovery of three new hare lagoviruses reveals unexplored viral diversity in this genus.

Our knowledge of mammalian viruses has been strongly skewed toward those that cause disease in humans and animals. However, recent metagenomic studies indicate that most apparently healthy organisms carry viruses, and that these seemingly benign viruses may comprise the bulk of virus diversity. The bias toward studying viruses associated with overt disease is apparent in the lagoviruses (family Caliciviridae) that infect rabbits and hares: although most attention has been directed toward the highly pathogenic members of this genus—rabbit haemorrhagic disease virus and European brown hare syndrome virus—a number of benign lagoviruses have also been identified. To determine whether wild European brown hares in Australia might also carry undetected benign viruses, we used a meta-transcriptomics approach to explore the gut and liver RNA viromes of these invasive animals. This led to the discovery of three new lagoviruses. While one was only detected in a single hare, the other two viruses were detected in 20 per cent of all animals tested. All three viruses were most closely related to other hare lagoviruses, but were phylogenetically distinct from both known viruses and from each other, indicating that lagoviruses have circulated for longer than previously assumed. Their evolution was also characterised by complex recombination events. Mapping mutations onto the lagovirus phylogeny revealed no amino acid changes that were consistently associated with virulence phenotype. Overall, our study points to extensive unsampled diversity in this genus, such that additional metagenomic studies are needed to fill gaps in the lagovirus phylogeny and better understand the evolutionary history of this important group of mammalian viruses.

Bilateral Brown Sendromunda Spontan İyileşme Gösteren ve Cerrahi Uygulanan Hastalardaki Klinik Faktörler

Bilateral Brown Sendromunda Spontan İyileşme Gösteren ve Cerrahi Uygulanan Hastalardaki Klinik Faktörler

First Complete Genome Sequence of a Hare Calicivirus Strain Isolated from Lepus europaeus.

The first full-genome sequence of a hare calicivirus (HaCV), recently characterized as a novel member of the Caliciviridae, is described. This presumed nonpathogenic lagovirus is 7,433 nucleotides long, shows the same genomic organization as that of other lagoviruses, and has the highest nucleotide identity (79%) with pathogenic European brown hare syndrome viruses.

Overcoming species barriers: an outbreak of Lagovirus europaeus GI.2/RHDV2 in an isolated population of mountain hares (Lepus timidus)

BackgroundPrior to 2010, the lagoviruses that cause rabbit hemorrhagic disease (RHD) in European rabbits (Oryctolagus cuniculus) and European brown hare syndrome (EBHS) in hares (Lepus spp.) were generally genus-specific. However, in 2010, rabbit hemorrhagic disease virus 2 (RHDV2), also known as Lagovirus europaeus GI.2, emerged and had the distinguishing ability to cause disease in both rabbits and certain hare species. The mountain hare (Lepus timidus) is native to Sweden and is susceptible to European brown hare syndrome virus (EBHSV), also called Lagovirus europaeus GII.1. While most mountain hare populations are found on the mainland, isolated populations also exist on islands. Here we investigate a mortality event in mountain hares on the small island of Hallands Väderö where other leporid species, including rabbits, are absent.ResultsPost-mortem and microscopic examination of three mountain hare carcasses collected from early November 2016 to mid-March 2017 revealed acute hepatic necrosis consistent with pathogenic lagovirus infection. Using immunohistochemistry, lagoviral capsid antigen was visualized within lesions, both in hepatocytes and macrophages. Genotyping and immunotyping of the virus independently confirmed infection with L. europaeus GI.2, not GII.1. Phylogenetic analyses of the vp60 gene grouped mountain hare strains together with a rabbit strain from an outbreak of GI.2 in July 2016, collected approximately 50 km away on the mainland.ConclusionsThis is the first documented infection of GI.2 in mountain hares and further expands the host range of GI.2. Lesions and tissue distribution mimic those of GII.1 in mountain hares. The virus was most likely initially introduced from a concurrent, large-scale GI.2 outbreak in rabbits on the adjacent mainland, providing another example of how readily this virus can spread. The mortality event in mountain hares lasted for at least 4.5 months in the absence of rabbits, which would have required virus circulation among mountain hares, environmental persistence and/or multiple introductions. This marks the fourth Lepus species that can succumb to GI.2 infection, suggesting that susceptibility to GI.2 may be common in Lepus species. Measures to minimize the spread of GI.2 to vulnerable Lepus populations therefore are prudent.

Brown Bowel Syndrome Presenting as a Small Bowel Obstruction

Abstract Brown bowel syndrome (BBS) is a rare condition characterized by lipofuscin deposition in the muscularis propria of the bowel. It is associated with vitamin E deficiency and malabsorptive syndromes. We report a case of BBS in a patient with previously undiagnosed primary intestinal lymphangiectasia. The patient had a longstanding history of malnutrition as well as bilateral lower extremity lymphedema and chylous ascites. He presented to our hospital with small bowel obstruction (SBO). Initial magnetic resonance enterography revealed a dilated duodenum, which when biopsied revealed only lymphangiectasia and lymphedema. He was managed conservatively and discharged following resolution of the obstruction. The patient presented again 1 month later with SBO that failed to resolve with conservative treatment. Subsequent exploratory laparotomy discovered a portion of pale, edematous, aperistaltic jejunum. The specimen was resected, and pathologic examination revealed brown granular pigment deposition within the muscularis propria, which was highlighted by periodic acid–Schiff and lipofuscin stains, diagnostic of BBS. Nutritional workup revealed multiple deficiencies. The patient's clinical findings and history were suggestive of primary intestinal lymphangiectasia with resulting nutritional deficiencies leading to BBS. Outpatient follow-up after recovery revealed an elevated 24-hour fecal α1 antitrypsin, which supports this diagnosis. The patient has since improved following adherence to a low-fat diet. The combined clinical, radiographic, and pathologic findings in this case allowed for the resolution of this complex diagnostic dilemma. Although rare, BBS remains an important consideration in the differential diagnosis of a patient with SBO and history of chylous ascites and should be viewed as a symptom of underlying treatable nutritional deficiency.

Melanosis? Think Again: A Case of Pseudomelanosis duodeni and Jejunal Hemangiomas

Melanosis coli is a common cause of hyperpigmented lesions in the GI tract that typically result from laxative overuse. Herein we describe a case of pseudomelanosis duodeni and jejunal hemangiomas discovered after push enteroscopy for an un-localized GI bleed. Though similar in appearance, pseudomelanosis is actually due to deposition of hemosiderin in lamina propria (LP) macrophages. An 83-year-old Asian female presented with recurrent hospitalizations for melena and severe anemia. Past medical history included transfusion-dependent iron deficiency anemia from chronic GI bleeding, hypertension, diabetes, heart failure, and atrial fibrillation not on anticoagulation. Her initial workup began in 2014 and EGD demonstrated erosive gastropathy with negative H. pylori. Colonoscopy demonstrated three 3-5 mm polyps and diverticulosis but no signs of acute bleeding. Repeat hospitalization for anemia with EGD in 2015 showed no source of bleeding. Colonoscopy showed two 5-8 mm polyps and diverticulosis but no bleeding. Outpatient capsule endoscopy in 2015 was unrevealing. Repeat hospitalization in April 2018 with colonoscopy revealed melena from the ileum and several small polyps in the cecal region. Push-enteroscopy revealed dark speckled pigmentation in the duodenum and jejunum. Two bleeding hemangiomas in the jejunum were found and ablated with APC then clipped. Duodenal biopsies demonstrated hemosiderin deposition within the LP macrophages consistent with a diagnosis of pseudomelanosis duodeni. Pseudomelanosis duodeni is characterized by the presence of brownish or blackish pigmentation in duodenal mucosa. It is associated with various medical conditions including, diabetes, renal disease, GI bleeding, hypertension, heart failure, oral iron intake, and use of erythropoiesis-stimulating agents. Histologic differential includes hemochromatosis, brown bowel syndrome, and malignant melanoma. Pseudomelanosis duodeni has histology notable for iron sulfide and hemosiderin deposits in the LP macrophages. This is in contrast to the accumulation of lipofuscin in the LP macrophages due to laxative overuse seen in melanosis coli. Interestingly, the hyperpigmentation made it easier to identify the jejunal hemangiomas; in a similar fashion to melanosis coli making it easier to identify polyps. Although this disease is quite rare and benign, it warrants tissue biopsy to rule out other worrisome etiologies.2546_A Figure 1. Dark speckled pigmentation scattered throughout the small bowel.2546_B Figure 2. Hemangiomas in the jejunum (blue/white arrows). Right image shows bleeding hemangioma (blue/white arrow).2546_C Figure 3. Left image demonstrates mucosal changes after APC ablation of bleeding hemangiomas (white/blue arrows). Right image demonstrates clips placed (red/white arrow) after APC ablation.

Consecutive superior oblique palsy after adjustable suture spacer surgery for Brown syndrome: incidence and predicting risk

To determine the incidence of and to identify characteristics predicting significant superior oblique palsy (SOP) after adjustable superior oblique suture spacer surgery for treatment of Brown syndrome. The medical records of patients treated for unilateral Brown syndrome with adjustable suture spacers (2005-2016) were reviewed to identify possible association of age at surgery, spacer length, surgeon performing procedure, severity of Brown syndrome, preoperative hypotropia in primary position and affected side gaze, and reduction in Brown restriction on postoperative superior oblique function. "Good" postoperative superior oblique function was defined as absence of hypertropia and diplopia in primary position and no more than intermittent diplopia in downgaze comfortably fused with ≤4Δ base-down or head tilt of <10°. Presence of postoperative hypertropia in primary position with increase in downgaze met criteria for significant SOP. Postoperative Brown restriction of≤-2 indicated resolution of Brown syndrome. Median age at surgery was 59months, interquartile range (IQR) was 32-82months, and median spacer length was 6mm (range, 2-7mm) for 19 included patients. Preoperative median hypotropia was 9Δ (IQR, 0Δ-12Δ) in primary position and 18Δ (IQR, 5Δ-22Δ) in affected side gaze. Of 19 patients, 16 (84%) achieved sufficient resolution of Brown syndrome, but 6 (32%) developed significant SOP. Modest preoperative hypotropia in affected side gaze was the only predictor of significant SOP (likelihood ratio test=7.11; P=0.008). Logistic regression modeling enabled estimation of risk of significant SOP based on preoperative side gaze hypotropia. Suture spacer surgery can result in significant SOP. Risk may be predicted by magnitude of preoperative side gaze hypotropia.

First complete genome sequence of a European non-pathogenic rabbit calicivirus (lagovirus GI.3)

We report the full genome sequence of the non-pathogenic rabbit lagovirus Lagovirus europaeus/GI.3/O cun/FR/2006/06-11 (GI.3/06-11), collected from a healthy French domestic rabbit in 2006, and initially described as 06-11 strain. The sequence reveals a genomic organization similar to lagoviruses. It was 7,436 bases long and contained two open reading frames (ORF). A dipeptide variation at the potential p23/2C-like helicase cleavage site (EE instead of ED) was observed, a feature only shared with non-recombinant pathogenic lagoviruses in GI.2 and with two European brown hare syndrome viruses (EBHSV) collected in 1982 in Sweden. GI.3/06-11 has only one initiation codon at the beginning of the ORF2 like the avirulent Italian rabbit calicivirus (RCV) and EBHSV. Previous genetic analyses based on the capsid gene sequences showed that GI.3/06-11 was closer to the RCV and pathogenic lagoviruses GI.1 strains than other lagoviruses. This study, by revealing that GI.3/06-11 genome sequence significantly clustered with pathogenic GI.2 strains, gives prominence of new genetic relationship among lagoviruses and should contribute to understand the emergence of pathogenic strains.

A Case Report of Progressive Brown Syndrome?

Aim:To report an unusual case of progressive Brown syndrome and the successful surgical treatment.Methods:A 42-year-old male with a documented 14-year history of progressive Brown syndrome is presented. To improve diplopia symptoms an ipsilateral superior oblique (SO) 7 mm silicone tendon spacer and a contralateral 3 mm superior rectus (SR) recession were performed.Results:The surgical procedure was successful in reducing the primary position deviation (preoperative 30ΔLHoT 7ΔLXT; one month post surgery 3ΔLHo; one year post surgery 7-8ΔLHo 1-2ΔE) and eliminating the abnormal head posture (AHP). Diplopia was only reported on elevation and depression following surgery.Conclusion:This case of Brown syndrome is unusual as it was progressive and had a documented history over a number of years prior to surgery. Surgical treatment of Brown syndrome is rarely required, but in this case was successful in improving the patient’s diplopia and AHP.