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Year : 2020  |  Volume : 68  |  Issue : 4  |  Page : 641-642

An unusual case of orbital myositis in a patient with HLA B-27-associated uveitis

1 Department of Uvea, Reconstructive and Aesthetic Services, Medical and Vision Research Foundations, Sankara Nethralaya, Chennai, Tamil Nadu, India
2 Department of Orbit, Oculoplasty, Reconstructive and Aesthetic Services, Medical and Vision Research Foundations, Sankara Nethralaya, Chennai, Tamil Nadu, India
3 Department of Radiology, VRR Scans, Chennai, Tamil Nadu, India

Date of Submission23-Jul-2019
Date of Acceptance06-Oct-2019
Date of Web Publication16-Mar-2020

Correspondence Address:
Dr. Parthopratim Dutta Majumder
Department of Uvea, Sankara Nethralaya, 18, College Road, Nungambakkam, Chennai - 600 006, Tamil Nadu
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_1360_19

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Keywords: Acute anterior uveitis, diplopia, HLA B-27, orbital myositis, uveitis

How to cite this article:
Dutta Majumder P, Koka K, Noronha V. An unusual case of orbital myositis in a patient with HLA B-27-associated uveitis. Indian J Ophthalmol 2020;68:641-2

How to cite this URL:
Dutta Majumder P, Koka K, Noronha V. An unusual case of orbital myositis in a patient with HLA B-27-associated uveitis. Indian J Ophthalmol [serial online] 2020 [cited 2020 Apr 2];68:641-2. Available from: http://www.ijo.in/text.asp?2020/68/4/641/280706

A 41-year-old lady presented to us with complaints of redness, ocular pain in left eye, and seeing double for the last 3 months. She was diagnosed as acute anterior uveitis (AAU) and was started on topical corticosteroid, cycloplegic elsewhere. Binocular diplopia which was initially in all gazes had reduced and currently she complained of persistent diplopia mainly in up gaze. She complained of recurrent attacks of redness with ocular pain in left eye for last 1 year, but denied any history of diplopia. On enquiry, she also complained of mild backache for past six months. Her past surgical history included laparoscopic cholecystectomy with metal clips done eight years back.

On examination, her best corrected visual acuity in both eyes was 6/6. Slit-lamp examination of the right eye was unremarkable. Slit-lamp examination of the left eye revealed cells 0.5+, flare 1+, and pigments over anterior lens capsule suggestive of broken synechiae [Figure 1]. Ocular motility examination of the right eye was within normal limit whereas left eye showed limited abduction [Figure 2]. Fundus examination of both eyes was normal. She was investigated extensively to determine the possible etiology. Her HLA typing was positive for HLA B-27. Her other investigations which also included thyroid function tests were all normal. An orbital computed tomography (CT) scan suggested mild to moderate enlargement of left lateral and inferior rectus muscles suggestive of myositis [Figure 3]a and [Figure 3]b. She was also evaluated by a rheumatologist and started on oral corticosteroid (60 mg/day) and oral methotrexate (15 mg/week). She was seen again after two months. Slit-lamp examination of the left eye revealed a quiet anterior chamber and she was symptomatically much better. She was advised to continue the same treatment in tapering schedule. She was again seen after four months of initial presentation. Slit-lamp examination of the both eyes revealed quiet anterior chamber without any ciliary congestion. She had marked reduction in diplopia and CT scan of the orbit showed resolution of the muscle thickness [Figure 3]c and [Figure 3]d.
Figure 1: Slit-lamp photograph of the left eye showing pigments over anterior lens capsule

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Figure 2: Diplopia charting done with red filter in right eye and green filter in left eye revealed fusion in all gazes except upper gazes and levo-version

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Figure 3: (a) and (b) Pretreatment computed tomography scan, axial, and coronal sections showing mild to moderate enlargement of left lateral and inferior rectus muscle. (c) and (d) Four-month posttreatment images showing reduction in size of the left lateral and inferior rectus muscle corresponding clinically with decrease in amount of diplopia

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Among the vast spectrum of ocular manifestations in HLA-B27-associated diseases, AAU remains the most common. Our patient had myositis and an extensive investigation ruled out all other possible causes of myositis. Myositis secondary to HLA-B27-associated uveitis is extremely rare. Sachdeva et al.[1] reported a case of enthesitis in a 29-year-old female with HLA-B-27-associated spondyloarthropathy. Unlike our patient, the patient had recurrent orbital pain, periorbital edema, and proptosis, in conjunction with severe sacroiliitis and peripheral arthritis. Our patient presented with painful diplopia with history of recurrent attacks of acute anterior uveitis and had milder form of spondyloarthropathy as suggested by rheumatologist. We did not observe signs and symptoms of orbital involvement such as orbital pain, proptosis, or chemosis. CT scan to evaluate her diplopia revealed the myositis. Our patient presented to us two months after the acute episode of anterior uveitis. Thus, it will be difficult to speculate the initial inflammatory episodes of events. Her 2-month old medical records showed lid edema, restriction of the eye movement, and AAU. Lid edema and restriction of the eye movement can be suggestive of orbital myositis; however, detailed evaluation of the patient was not done or documented. Several systemic rheumatic diseases such as rheumatoid arthritis, systemic lupus, and erythematosus have been reported in association with orbital myositis.[2] Another report describes a case of orbital myositis in a 3-year-old female child with juvenile psoriatic arthritis, but child was tested negative for HLA-B27.[3] Our report suggests that orbital myositis can be a rare association of HLA-B27-associated ocular disease and should be properly evaluated in patients developing painful diplopia.

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.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Sachdeva A, Kramer N, Rosenstein ED. Orbital inflammatory disease: unusual presentation of enthesitis in an HLA-B27 spondyloarthropathy. Ocul Immunol Inflamm 2012;20:468-70.  Back to cited text no. 1
Weinstein GS, Dresner SC, Slamovits TL, Kennerdell JS. Acute and subacute orbital myositis. Am J Ophthalmol 1983;96:209-17.  Back to cited text no. 2
Ajitsaria R, Dale R, Ferguson V, Mayou S, Cavanagh N. Psoriasis, psoriatic arthropathy and relapsing orbital myositis. Clin Exp Dermatol 2001;26:274-5.  Back to cited text no. 3


  [Figure 1], [Figure 2], [Figure 3]


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