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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 3  |  Page : 580-581

A case of cyclical Brown's syndrome


Departement of Pediatric Ophthalmology and Strabismus, Aravind Eye Hospital, Madurai, Tamil Nadu, India

Date of Submission23-Jul-2020
Date of Acceptance27-Feb-2021
Date of Web Publication02-Jul-2021

Correspondence Address:
Dr. Shashikant Shetty
Departement of Pediatric Ophthalmology and Strabismus, Aravind Eye Hospital, Madurai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2365_20

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  Abstract 


A 5-year-old girl presented with limitation of elevation in adduction in her left eye. She had history of intermittent deviation in the left eye for the past 2 months. Parents gave a characteristic history that she had an obvious deviation when she woke up in the morning and resolved as the day progressed. Neuroimaging and ocular examination was unremarkable. Only one such case of intermittent Brown's syndrome has been reported in the past in a patient with history of maxillary sinusitis. Herein, we present a case that shows a unique presentation of an intermittent heterotropia without any systemic associations.

Keywords: Brown's syndrome, intermittent Brown's syndrome, ocular motility deficit, superior oblique


How to cite this article:
Ganatra S, Shetty S, Prajna M, Chauhan N, Vijayalakshmi P. A case of cyclical Brown's syndrome. Indian J Ophthalmol Case Rep 2021;1:580-1

How to cite this URL:
Ganatra S, Shetty S, Prajna M, Chauhan N, Vijayalakshmi P. A case of cyclical Brown's syndrome. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Jul 26];1:580-1. Available from: https://www.ijoreports.in/text.asp?2021/1/3/580/319992



Brown's syndrome is a disorder of ocular motility described in 1950 by Harold W. Brown characterized by restriction of elevation in adduction. It can be congenital or acquired due to inflammation or trauma to the trochlear region. Neuroimaging, orbital imaging and relevant blood investigations should be done to find out the cause. Herein, we report an interesting case of intermittent Brown's syndrome.


  Case Report Top


A 5-year-old girl was referred to our pediatric ophthalmology department by her primary care giver as a case of strabismus. The parents gave a history of noticing deviation in the child's left eye, on and off, for the past 2 months. There was no history of trauma or systemic illness. Her old photographs were unremarkable. On examination, her uncorrected visual acuity (UCVA) was 20/20 in both eyes. Bilateral anterior and posterior segment examination was unremarkable. Ocular movement examination did not reveal any obvious limitation [Figure 1]. Cover test revealed no deviation. The parents were reassured and were informed to report in case of any further symptoms.
Figure 1: Ocular movement examination did not reveal any obvious limitation

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The child was brought to us by her father a week later with complaints of deviation in left eye. He gave the history that the deviation appeared on waking up and gradually resolved as the day progressed. Her UCVA was maintained at 20/20. On examination, she had a left face turn and a small chin elevation. The corneal reflex showed a small angle hypotropia in the left eye. Cover tests showed left hypotropia. There was limited elevation in adduction (–4) in her left eye with associated palpebral fissure widening on adduction [Figure 2]. The extra-ocular movements in her right eye were unremarkable. The child was re-examined the same evening and there was improvement in the deviation. A diagnosis of acquired intermittent Brown's syndrome of the left eye was made. We advised MRI Brain, thyroid function tests, complete blood count, erythrocyte sedimentation rate, C-reactive protein, and rheumatoid factor. All investigations were unremarkable. Coronal section of T1 W MRI revealed a normal study with no abnormality in the trochlear region. No sinus mucosal thickening was noted [Figure 3] After 10 days, the child was brought to us again and examination revealed no obvious deviation and extra-ocular movements were full in both eyes. The child's father however gave the history that the child continues to have intermittent deviation. We reassured the father and advised periodic review checkup to monitor for progression of the condition. The patient was advised to review every 4 months as she is in the amblyogenic age group.
Figure 2: There was limited elevation in adduction (-4) in her left eye with associated palpebral fissure widening on adduction

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Figure 3: Coronal section of T1 W MRI revealed a normal study with no abnormality in the trochlear region

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  Discussion Top


We report a case of Intermittent Brown's syndrome which appears after the patient wakes up from sleep and resolves spontaneously within a few hours. This phenomenon is postulated to be occurring due to accumulation of fluid that occurs in the inflamed tissues during sleep, a mechanism akin to that in rheumatoid arthritis. The edema decreases as the day progresses because the fluid is returned to circulation by muscle action via the lymphatics and venules.[1] In our patient, test for rheumatoid factor was negative.

Acquired Brown's syndrome is caused due to trauma, sinusitis, after orbital, retinal, or sinus surgery, post-operative after superior oblique surgery, and inflammatory conditions such as juvenile rheumatoid arthritis, SLE, hypogammaglobulinemia, and post-streptococcal reactive arthritis.[2],[3] To our knowledge, only one such case has been reported wherein the patient had similar complaints but had a positive history of maxillary sinusitis.[4] Pan sinusitis and most commonly ethmoidal sinusitis can also be a cause of acquired Brown's syndrome.

Neurocysticercosis is a rare but an important cause of acquired Brown's syndrome in children. Other rare causes which could be considered are orbital masses, nasal masses, and thyroid ophthalmopathy. In patients having trauma to the trochlear region and post-surgery, restriction of movement of the tendon is caused by scarring and fibrosis between the trochlea and the superior oblique tendon.[2] Spontaneous resolution of limitation in adduction in Brown's syndrome has been previously described. Costenbader et al.[3] reported about the spontaneous resolution of Brown syndrome in a 6½-year-old boy after a 3-year follow-up. Adler[5] also reported about the spontaneous resolution of Brown syndrome in a 7-year-old boy after a 3-year follow-up. Leone et al.[6] too reported about the spontaneous resolution of Brown syndrome in a 21-year-old man that was attributed to daily eye exercise and was also associated with an audible click. Can et al.[4] reported a case of a 15-year-old boy having limited elevation in adduction which appeared as he woke up from sleep and eventually resolved spontaneously during the day, the patient had a positive history of maxillary sinusitis.


  Conclusion Top


To our knowledge and after a through literature search, we believe that such a case of intermittent Brown's syndrome without a definite cause has not been reported in the pediatric population. The importance lies in the regular follow-up of this child to monitor the deviation in order to avoid amblyopia in case the deviation becomes constant. We want to emphasize the need and role of systemic evaluation in a child presenting with acquired Brown's syndrome, since a number of systemic causes can give rise to this condition. Management in our case involved masterly inactivity.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Harris ED Jr. Clinical features of rheumatoid arthritis. In: Kelly WN, Harris ED Jr, Ruddy S, Sledge CB, editors. Textbook of Rheumatology. 4th ed. Philadelphia, PA: Saunders; 1993. p. 874-8.  Back to cited text no. 1
    
2.
Pronk AE, Demaerel PM, Casteels IK. A 5-year-old boy with acute intermittent acquired Brown's syndrome. Bull Soc Belge Ophtalmol 2010;316:21-6.  Back to cited text no. 2
    
3.
Costenbader FD, Albert DG. Spontaneous regression of pseudoparalysis of the inferior oblique muscle. AMA Arch Ophthalmol 1958;59:607-8.  Back to cited text no. 3
    
4.
Can I, Yarangümeli A, Kural G. Brown's syndrome with cyclic characteristic: Case report and review of physiopathologic mechanism. J Pediatr Ophthalmol Strabismus 1995;32:243-7.  Back to cited text no. 4
    
5.
Adler FH. Spontaneous recovery in a case of superior oblique sheath syndrome of Brown. Arch Ophthalmol 1959;61:1006-7.  Back to cited text no. 5
    
6.
Leone CR Jr, Leone RT. Spontaneous cure of congenital Brown's syndrome. Am J Ophthalmol 1986;102:542-3.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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