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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 520-521

The eye in lock-down: An unusual acquired restrictive strabismus


1 Jasti V Ramanamma Children's Eye Care Centre, L.V. Prasad Eye Institute, Hyderabad, India
2 Ophthalmic Plastic Surgery Service, L.V. Prasad Eye Institute, Hyderabad, India
3 Department of Pediatric Ophthalmology, Children's Hospital at Westmead, Sydney, Australia
4 Department of Neuro-Radiology, CitiNeuro Centre, Hyderabad, India

Date of Submission10-Aug-2021
Date of Acceptance19-Oct-2021
Date of Web Publication13-Apr-2022

Correspondence Address:
Manjushree Bhate
Associate Ophthalmologist, L. V. Prasad Eye Institute, Hyderabad - 500 034, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2076_21

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  Abstract 


We report a rare case of a 3-year-old female child with a severe degree of hypotropia and esotropia of the left eye secondary to presumed myositis from unknown etiology treated with systemic steroids. A magnetic resonance imaging (MRI) of the orbit revealed a bulky left inferior rectus muscle. With a working diagnosis of inferior rectus contracture, and presumed dense amblyopia in the left eye, a free tenotomy of the left inferior rectus muscle along with recession of the left medial rectus restored orthotropia from the locked-down position.

Keywords: Esotropia, free tenotomy, hypotropia, restrictive strabismus


How to cite this article:
Bhate M, Naik MN, Martin FJ, Varma R. The eye in lock-down: An unusual acquired restrictive strabismus. Indian J Ophthalmol Case Rep 2022;2:520-1

How to cite this URL:
Bhate M, Naik MN, Martin FJ, Varma R. The eye in lock-down: An unusual acquired restrictive strabismus. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 26];2:520-1. Available from: https://www.ijoreports.in/text.asp?2022/2/2/520/342923



Acquired restrictive strabismus in a young child poses management challenges. We present an unusual case in a patient who presented with a profound degree of hypotropia and esotropia of the left eye from unknown etiology after several months of onset. A challenging strabismus surgical approach restored the ocular alignment.


  Case Report Top


A 3-year-old female child was brought to the strabismus clinic at L.V. Prasad Eye Institute Hyderabad with the complaint of sudden onset inward and downward deviation of the left eye 10 months prior to presentation. There was no history of trauma, fever, or any illness in the recent past. Old photographs of the child did not reveal any deviation and the eyes appeared orthotropic [Figure 1]a. On examination, an extreme esotropia and hypotropia of the left eye was noted at the initial presentation [Figure 1]b. There was no ocular motility in the left eye and the eye was locked in a “down and in” position. The right eye motility was full. The anterior and posterior segment examination was unremarkable in both eyes. MRI of the orbit revealed increased bulk and signal intensity of the posterior two-thirds of the left inferior rectus muscle [Figure 3]a,[Figure 3]b,[Figure 3]c. Thyroid serology was performed and was found to be normal.
Figure 1: (a) Orthotropia at 1 year of age, (b) Extreme hypotropia and esotropia at presentation, (c) Worsening of deviation over the months

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Figure 2: Primary position at post-operative follow up visit 3 months after strabismus surgery

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Figure 3: (a-c) Coronal and sagittal images showing the bulky left inferior rectus muscle (d) Normal size right inferior rectus muscle

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The child was treated with a course of systemic steroids for a presumed diagnosis of myositis, without any improvement, and a repeat MRI after 2 months showed persistent thickening of the inferior rectus muscle. Further consultation was delayed by 8 months due to the COVID-19 pandemic related lockdown, following which the esotropia and hypotropia had worsened [Figure 1]c, with no significant interval change in the bulk and signal intensity of the left inferior rectus on repeat MRI orbit. Surgical intervention was required considering the extreme strabismus and the possibility of amblyopia.

Strabismus surgery on the left eye was performed under general anesthesia. Following a large limbal peritomy, the medial rectus was approached first by gently hooking the muscle, minimizing pull on the muscle. The muscle was found to be extremely tight on attempted forced duction testing (FDT). A large medial rectus recession of 10 mm from the insertion on a hangback suture was carried out. The extremely tight inferior rectus muscle was then approached with the assistance of an oculoplastic surgeon via inferior forniceal incision and a free tenotomy in the mid-orbit was performed. The FDT performed at the end of the surgery confirmed no restriction with full range of rotation of the globe. An amniotic membrane graft was placed in the inferior fornix. Postoperative, there was a significant improvement in the ocular position and amblyopia therapy in the form of patching was commenced. At the 3-month follow-up visit, the child was orthotropic on cover test for near [Figure 2] and had full range of adduction with −1 abduction and −3 depression of the left eye.


  Discussion Top


Acquired extraocular muscle fibrosis is rare, and can be caused by Graves' disease, trauma, myositis, or abnormal bands.[1],[2],[3],[4] Orbital myositis has been described as a subgroup of idiopathic orbital inflammatory disease. In children, orbital myositis has been reported following upper respiratory infection or pharyngitis while a delayed onset myositis associated with bacterial or viral disease likely suggests an immune-mediated process.[5] It has also been reported following Lyme disease,[6] cysticercosis, and non-Hodgkin's lymphoma.[7],[8] A lack of severe pain in children has been reported.[5] One of the earliest reports by Paolillo et al.[1] describes an extreme infra-duction and inability to elevate the left eye in a 4-month-old infant. They discovered a thin taut band replacing the inferior rectus muscle. Souza-Dias and colleagues have reported three cases with isolated acquired muscle restrictions involving the superior and medial recti in different subjects. Two of their subjects had serological tests for Graves' disease which were reported as normal, none were treated with systemic steroids and all of them underwent strabismus surgery with variable results.[5] The authors suggested myositis as the possible etiology in all these cases due to the progressive course and the imaging findings. The absence of severe pain because of the subacute nature of presentation.[5]

Slavin and Glaser[9] reported 6 cases of orbital myositis, one of which was a child presenting after a flu-like illness with proptosis and ophthalmoplegia and noted to have an enlargement of the superior and medial recti which resolved completely following a course of oral steroids. Pollard reported on a series of patients presenting with acute rectus muscle palsy as a result of orbital myositis, who recovered completely following course of oral steroids and mention the inferior rectus muscle to be least involved in orbital myositis as opposed to Graves' disease.[2]

Our patient is likely to have had the fibrosis and contracture of the inferior rectus muscle following presumed myositis, leading to the restriction which resulted in a profound degree of hypotropia with esotropia and absent ocular motility.

An MRI finding of an irregular contour and shortened right superior rectus muscle was noted in an acquired oculomotor fibrosis presenting in an infant with an extreme upward and lateral deviation of the right eye by Souza-Dias et al.[4] The child underwent a free tenotomy of the superior rectus with recession of the lateral rectus.[4] Our patient had an involvement of the inferior rectus in the left eye.


  Conclusion Top


Our patient's strabismus did not reverse with 3 months of systemic steroids possibly due to late presentation in the course of the disease. Successful ocular alignment was restored in this patient after the strabismus surgery.

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

Hyderabad Eye research foundation.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Paolillo RD, Burch OG, Torchia RT. Infantile contracture of inferior rectus muscle with resultant mechanical hypotropia. Am J Ophthalmol 1969;68:1057-60.  Back to cited text no. 1
    
2.
Pollard ZF. Acute rectus muscle palsy in children as a result of orbital myositis. J Pediatr 1996;128:230-3.  Back to cited text no. 2
    
3.
Chan W, Wong GWK, Fan DSP. Ophthalmopathy in childhood graves disease. Br J Ophthalmol 2002;86:740-2.  Back to cited text no. 3
    
4.
Souza-Dias C, Goldchmit M, Uesugu CF. Acquired oculomotor muscle fibrosis in infant Arq Bras Oftalmol 2011;74:211-3.  Back to cited text no. 4
    
5.
Souza-Dias C, Scott AB, Wang AH. Progressive restrictive strabismus acquired in infancy. Br J Ophthalmol 2005;89:986-7.  Back to cited text no. 5
    
6.
Seidenberg KB, Leib ML. Orbital myositis with Lyme disease. Am J Ophthalmol 1990;109:13-5.  Back to cited text no. 6
    
7.
Stewart R, Salmon JF, Murray AD, Sperryn C. Cysticercosis as a cause of severe medial rectus muscle myositis. Am J Ophthalmol 1993;116:510-1.  Back to cited text no. 7
    
8.
Harris, GH, Murphy M, Schmidt EW, Hanson GA, Dotson RM. Orbital myositis as a paraneoplastic syndrome. Arch Ophthalmol 1994;112:380-6.  Back to cited text no. 8
    
9.
Slavin ML, Glaser JS. Idiopathic orbital myositis. Arch Ophthalmol 1982;100:1261-5.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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