Bilateral internuclear ophthalmoplegia developing as a manifestation of dengue fever and treatment by modified Nishida procedure: A case report and review of the literature

Neelam Pawar; Meenakshi Ravindran; S Padmavathy; Sabyasachi Chakrabarty; Anurag Gandhi

doi:10.4103/ijo.IJO_1669_21

CASE REPORT

Year : 2022 | Volume : 2 | Issue : 1 | Page : 195-197

Bilateral internuclear ophthalmoplegia developing as a manifestation of dengue fever and treatment by modified Nishida procedure: A case report and review of the literature

Neelam Pawar¹, Meenakshi Ravindran¹, S Padmavathy², Sabyasachi Chakrabarty¹, Anurag Gandhi¹
¹ Department of Pediatric Opthalmology and Strabismus, Aravind Eye Hospital, Tirunelveli, Tamil Nadu, India
² Department of Neurophthalmolgy, Aravind Eye Hospital, Tirunelveli, Tamil Nadu, India

Date of Submission	18-Jun-2021
Date of Acceptance	14-Aug-2021
Date of Web Publication	07-Jan-2022

Correspondence Address:
Dr. Neelam Pawar
Pediatric and Squint Services, Aravind Eye Hospital and Postgraduate Institute of Ophthalmology, Tirunelveli - 627 001, Tamil Nadu
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_1669_21

Abstract

Typical causes of internuclear ophthalmoplegia (INO) include demyelinating conditions, with inflammation, infection, and trauma being less common causes. Diplopia associated with INO severely impacts the patient's quality of life. Orthoptic treatment usually fails; therefore, appropriate surgical treatment should be considered. We report the unique case of a 25-year man with bilateral INO developing as a manifestation of dengue fever-related encephalitis and its successful treatment by modified Nishida procedure.

Keywords: Bilateral internuclear ophthalmoplegia, dengue fever, modified Nishida

How to cite this article:
Pawar N, Ravindran M, Padmavathy S, Chakrabarty S, Gandhi A. Bilateral internuclear ophthalmoplegia developing as a manifestation of dengue fever and treatment by modified Nishida procedure: A case report and review of the literature. Indian J Ophthalmol Case Rep 2022;2:195-7

How to cite this URL:
Pawar N, Ravindran M, Padmavathy S, Chakrabarty S, Gandhi A. Bilateral internuclear ophthalmoplegia developing as a manifestation of dengue fever and treatment by modified Nishida procedure: A case report and review of the literature. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Aug 14];2:195-7. Available from: https://www.ijoreports.in/text.asp?2022/2/1/195/334900

Demyelinating lesions are the most common cause of bilateral INO in the younger demographic. Other less common causes of INO include neurosyphilis, Arnold–Chiari malformation, head trauma, hydrocephalus, drug intoxication, and nutritional disorders such as Wernicke encephalopathy and pernicious anemia.^[1],[2]Among infectious causes AIDS, tuberculosis, syphilis, and cysticercosis have been reported for INO in the literature.^{[2],[3],[4],[5],[6]}

Case Report

A 26-year male presented to us with complaint of double vision since 12 months.He had history of altered sensorium, aphasia following fever 12 months back for which he was investigated and treated. His dengue IgM serum and cerebrospinal fluid (CSF) were positive during admission in a secondary center while HIV was nonreactive and Leptospira and Japanese encephalitis IgM were negative. His magnetic resonance imaging (MRI) brain had features suggestive of dengue encephalitis. He was diagnosed with dengue fever encephalitis with bilateral INO.

He presented to us later with complaint of persisting squinting and double vision since 12 months. On presentation, he had uncorrected visual acuity of 20/80 in both eyes and best-corrected visual acuity was 20/20. He denied any previous use of glasses. On dry retinoscopy, both eyes showed myopia of −3.00D, which disappeared after cycloplegic refraction. He was prescribed cyclopentolate 1% for this pseudomyopia.

Neuro-ophthalmic assessment in primary gaze revealed a large exotropia of ~90 prism diopters for distance and near fixation [Figure 1]. A horizontal abducting nystagmus and inability of the right eye to adduct with limitation of −5 was noted on left lateral gaze and adduction limitation −2 in the left eye with vertical nystagmus on right lateral gaze. The patient was able to converge on presentation of a near target despite the absence of voluntary adduction in both eye. Vertical movements were intact. The features were suggestive of long standing Bilateral INO secondary to dengue encephalitis. Fundus examination of both eye were within normal limits. Color vision was normal in both eyes.

Figure 1: Nine cardinal gazes showing bilateral limitation of adduction with large exotropia before surgery

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His previous MRI Axial FLAIR images showed bilateral symmetrical hyperintensities in bilateral cerebellar white matter, bilateral frontal lobes, and right thalamus and inferior pons with hemorrhages in the bilateral cerebellar hemisphere, right thalamus, bilateral frontal lobes, and inferior pons [Figure 2]a, [Figure 2]b, [Figure 2]c.

Figure 2: (a) showing hyperintensities in frontal lobes (b) showing hyperintensities in cerebellar white matter, and right thalamus and (c) showing hemorrhages in inferior pons (red circle)

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All examinations, investigations, and surgical intervention were done after his written informed consent. The transposition of a vertical rectus to medial rectus imposes the risk of anterior segment ischemia (ASI), and periosteal fixation of lateral rectus is unlikely to help in such large neurological exodeviation.

Intraoperatively, we found forced duction test mild tight in the right eye. Instead of the one-third muscle of original Nishida's width, we took 3/4^th of muscle and passed 8-mm nonabsorbable suture from original insertion in both superior rectus and inferior rectus. Nonabsorbable 5–0 braided polyester suture (Aurobond, Aurolab, Madurai, India) was passed 8 mm from muscle insertion in both superior rectus and inferior rectus including 3/4^th of muscle width similar to recession suture, and two ends were transposed 14 mm behind limbus near to medial rectus.

He underwent modified Nishida procedure in both eyes; right eye followed by left eye. After two weeks, he had a supramaximal recession of 12 mm of lateral rectus (LR) in RE. Bilateral transposition myopexy reduced the amount of exotropia from 90Base In (BI) (40° by Hirschberg) to 25–20 △ BI, and for the residual exotropia 12 mm LR recession in RE was performed. Finally, the patient had good cosmetical correction with residual exotropia of 10 △ BI and bilateral improvement of adduction by 25° with occasional diplopia in right lateral gaze [Figure 3].

Figure 3: Nine gazes showing improvement in abduction and angle of deviation after bilateral Nishida surgery and RE lateral rectus recession

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Discussion

In internuclear ophthalmoplegia, there is limited adduction in the eye ipsilateral to the affected medial longitudinal fasciculus, which ranges from mild limitation to severe restriction of adduction. There is an abducting nystagmus that usually lasts for a few beats.^{[1],[2],[3],[4],[5]} Demyelinating lesions are most common cause of bilateral INO in the younger demographic. Other less common causes of INO include infections, trauma, and vascular lesions.^{[2],[3],[4],[5],[6],[7]}

Ocular complications of dengue encephalitis, encephalopathy, and cerebellar syndromes have been reported.^[10] Though mononeuropathy, encephalopathy, transverse myelitis, polyradiculopathy, Guillain–Barre syndrome, oculomotor neuropathy, abducens nerve palsy, and optic neuropathy have been documented in the literature, bilateral INO has not been reported as a dengue-related complication.^[10]

Thalamic involvement with positive IgM in CSF for dengue virus is a key diagnostic feature. The MRI features were strongly suggestive of dengue encephalitis with hyperintensities in bilateral cerebellar hemispheres, bilateral frontal lobes, right thalamus, inferior pons, and hemorrhages in the frontal lobe, right thalamus, and pons, which led to INO in this case.

This case is unique in the aspect of infectious INO due to dengue encephalitis, which has not been reported earlier in the literature. Countries in which dengue poses challenges should be aware of this neurological complication of dengue fever. Diplopia associated with bilateral INO severely impacts the patient's quality of life; therefore, surgical treatment should be considered in young patients.

This patient had large-angle exotropia of 90 PD secondary to dengue-related INO. Our modified technique of transposition of the vertical recti (modified Nishida) successfully corrected large-angle exotropia and improved adduction in both eyes and obviates the risk of ASI.

Adams et al. reported successful surgical outcomes of three wall-eyed bilateral internuclear ophthalmoplegia (WEBINO) patients secondary to multiple sclerosis, who underwent bilateral resection and recession surgery to correct large-angle exotropia and diplopia.^[9]

Hall et al.^[8] performed two-stage unilateral recession-resection procedures in seven patients with vascular etiology; the remaining patients underwent bilateral LR recessions in eight cases of WEBINO with successful binocular outcomes.

Nathan et al.^[6] reported transposition surgery in five patients to be an effective option for surgical treatment of patients with internuclear ophthalmoplegia along with ipsilateral LR recession. Sajjadi et al.^[4] reported a single case of INO treated by transposition surgery with good surgical outcomes. All of the transposition surgeries involved tenotomies while our case had no split transposition surgery.

Due to the limited literature published on both recess–resect surgical intervention and transposition surgeries for the management of internuclear ophthalmoplegia, it is difficult to rely on a single surgical technique. Varying results have been reported from a very small number of patients with different etiology.^[8],[9],[10]

Conclusion

Our modified technique of transposition myopexy of the vertical recti successfully corrected large-angle exotropia and improved adduction without limiting abduction. Functional results can be achieved even in longstanding dengue-related INO cases.

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

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