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   Table of Contents      
CASE REPORT
Year : 2018  |  Volume : 5  |  Issue : 2  |  Page : 47-50

Lateral rectus paralysis: An emerging neuro-ophthalmic manifestation in dengue fever


1 Department of Ophthalmology, JSS Medical College and Hospital, JSS University, Mysore, Karnataka, India
2 Department of Medicine, JSS Medical College and Hospital, JSS University, Mysore, Karnataka, India

Date of Submission20-Aug-2018
Date of Acceptance09-Apr-2020
Date of Web Publication12-Jul-2020

Correspondence Address:
Dr. Prabhakar Srinivasapuram Krishnacharya
Department of Ophthalmology, JSS Medical College and Hospital, JSS University, Mysore - 570 004, Karnataka
India
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DOI: 10.4103/bijo.bijo_7_18

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  Abstract 


The purpose of presenting this case study is to find out the possible etiopathogenesis of lateral rectus (LR) palsy. A 17-year-old female patient presented with acute vomiting, diplopia, and bleeding per vagina for 3 days after 10 days of persistent fever. Restricted ocular movements in the right field of gaze suggested right LR paralysis, although diplopia charting performed by red and green glasses showed inconsistent results. Dilated fundoscopy examination revealed asymmetrical early papilledema. Reduced serum sodium levels less than 130 mEq/l with positive enzyme-linked immunosorbent assay NS1 antigen were found. Management included administration of intravenous fluids, inotropes, 20% mannitol, and 4 units of donor platelet transfusion. Symptomatic improvement with complete remission of diplopia was observed. This is the first case study that revealed the transient course of dengue-induced LR paralysis, the pathophysiology of which may possibly due to reduced serum sodium levels and/or raised intracranial pressure (ICP). The pathophysiology of raised ICP in dengue fever merits further research.

Keywords: Dengue viral infection, diplopia, electrolyte imbalance, lateral rectus paralysis


How to cite this article:
Krishnacharya PS, Kumar S, Jakka S, Maheshwari S. Lateral rectus paralysis: An emerging neuro-ophthalmic manifestation in dengue fever. Albasar Int J Ophthalmol 2018;5:47-50

How to cite this URL:
Krishnacharya PS, Kumar S, Jakka S, Maheshwari S. Lateral rectus paralysis: An emerging neuro-ophthalmic manifestation in dengue fever. Albasar Int J Ophthalmol [serial online] 2018 [cited 2021 Aug 3];5:47-50. Available from: https://www.bijojournal.org/text.asp?2018/5/2/47/289601




  Introduction Top


Dengue fever (DF) is an arthropod-borne public health problem caused by Flavivirus and transmitted by Aedes aegypti mosquitoes, which are both diurnal and container breeders typically present with varied manifestations in all age groups from classic DF (CDF) to dengue hemorrhagic fever (DHF) and dengue shock syndrome (DSS). CDF, or “breakbone fever,” is characterized by 3–14 days of high fever of acute onset following infected mosquito bite that presents with frontal headache, retro-orbital pain, myalgias, arthralgias, hemorrhagic manifestations, rash, and reduced white blood cell count. Few patients also might complain of anorexia and nausea. Acute symptoms when present usually last for about a week, nevertheless weakness, malaise, and anorexia might persist for several weeks. DHF presents with severe abdominal pain, persistent vomiting, fluctuating temperature (from fever to hypothermia), hemorrhagic manifestations, and higher cortical functional disorientation. DSS presents with restlessness, cold clammy skin, rapid weak pulse, and narrowing of pulse pressure.

The prevalence of DF is estimated at 100 million cases/year and worldwide, there are 2.5 billion people who are at risk.[1] In India, the risk of DF has shown an exponential increase in its incidence due to rapid urbanization, changing lifestyles, and deficient water management including improper water storage practices. Recent studies revealed the changing clinical profile of DF presenting with neurological and neuro-ophthalmic complications, although their exact incidence is uncertain. A previously published study reported the highest incidence of DF from Southeast Asia, India, and American tropical regions.[2]

Neurological manifestations such as encephalitis, encephalopathy, mononeuropathy, transverse myelitis, polyradiculopathy, and Guillain–Barre syndrome are associated with DF that showed wide variation in its incidence from 0.5% to 21%.[3],[4],[5],[6] However, now, widespread ocular manifestations extending from symptomatic visual disturbance to severe visual loss are being reported as cranial mononeuropathies such as isolated optic neuropathy and oculomotor nerve paralysis.[7],[8]

Diplopia caused by abducens nerve palsy as a neuro-ophthalmic complication following DF is rare that it had been reported thrice in the published medical literature. Currently, there are no reports on DF-induced lateral rectus (LR) paralysis due to raised intracranial pressure (ICP) and hyponatremia. The present case study describes the clinical course and pathogenesis of LR paralysis associated with a serologically proven case of dengue viral infection.


  Case Report Top


Here, we report the case of a 17-year-old young, otherwise normal female from the tropical region of South India, Mysore district, Karnataka state, with a history of acute vomiting, diplopia, and bleeding per vagina (PV) for the last 3 days, after 10 days of persistent fever. Informed written consent was obtained for publication purpose from the patient and her parents. On admission, pulse rate was 96 per min, regular, and of low volume and blood pressure was 80/60 mmHg with a 100.6°F temperature. A provisional diagnosis of DF was considered and managed with intravenous fluid bolus and inotropes to normalize the blood pressure, according to the WHO guidelines, and the patient was shifted to the intensive care unit when minimal improvement was noticed. Neurological examination was normal except for right LR paralysis for which magnetic resonance imaging (MRI) of the brain and magnetic resonance venogram were advised to rule out acute disseminated encephalomyelitis, cortical venous thrombosis, and other causes. Adjacent to the frontal horn of the lateral ventricle, a small periventricular cyst of 2 mm was observed. There were no signs of raised ICP on MRI, and the rest of the brain parenchyma showed normal signal intensity.

On the 3rd day of admission, visual acuity and near vision were 6/9 parts and N6, respectively. An automated refractometry reading of −0.50 sphere was found in both the eyes. Alternating esotropia of 15° was observed, and the primary deviation was equal to the secondary deviation on prism cover test. Restricted ocular motility in dextroversion, dextroelevation, and dextrodepression suggested right LR paralysis, although diplopia charting executed by red and green glasses showed occasional and inconsistent evidence of double vision [Figure 1] and [Figure 2]. Anterior segment examination was unremarkable for the cornea, lens, and iris, with briskly reacting 3-mm-sized pupil. Dilated fundoscopy by direct ophthalmoscope and Volk +90 D lens examination revealed hyperemia, an elevated optic disc with indistinct margins, a cup–disc ratio of 0.2 in the right eye, and obliteration of cup in the left eye that implied asymmetrical early papilledema. Retinal vessels, macular area, and retinal periphery were unremarkable in both the eyes.
Figure 1: Restricted abduction in the right field of gaze

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Figure 2: Globes in the primary gaze

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However, lumbar puncture was not performed as there was papilledema and there was no evidence of raised ICP on MRI.

Laboratory investigations showed hemoglobin (Hb) – 8.3 g/dl, red blood cell – 3.27 million/mm3, packed cell volume (PCV) – 26.1%, platelet count – 28,000/μl, thin-layer chromatography – 4480 cells/μl, serum glutamic oxaloacetic transaminase – 189 U/L, serum glutamic pyruvic transaminase – 80 U/L, erythrocyte sedimentation rate (ESR) – 70 mm/h by Wintrobe method, thyroid-stimulating hormone – 0.286 U/ml, total bilirubin – 3.28 mg/dl, direct bilirubin – 2.81 mg/dl, total proteins – 5.36 g/dl, albumin – 2.37 g/dl, albumin/globulin ratio – 0.9, and alkaline liver phosphatase – 72 IU/ml. Other investigatory results were as follows: electrolyte Na – 130 mEq/l, K – 4.7 mEq/l, Cl – 101 mEq/l, urea – 17 mg/dl, and creatinine – 0.7 mg/dl. High-sensitivity troponin T was 0.014 ng/ml and creatine kinase-MB was 1.41 ng/ml (COBAS E 411 Roche Hitachi Electrochemiluminescence). Malaria card test was negative, chemiluminescence immunoassay anti-HIV and anti-HbsAg was negative, enzyme-linked immunosorbent assay (ELISA) for Dengue NS1 antigen (Ag) by Panbio® Dengue Early ELISA was positive, and IgM for Leptospira by Panbio Leptospira ELISA and Weil–Felix slide agglutination by Plasmatech were negative.

Ultrasonography of the abdomen showed features of polyserositis with normal chest X-ray, electrocardiography, and echocardiography. Intravenous fluids and inotropes in addition to the administration of 4 units of random donor platelet transfusion were continued in view of bleeding PV and thrombocytopenia in the intensive care unit. The patient's general condition improved symptomatically after 48 h of management except for double vision. In view of the features of early papilledema, probably secondary to raised ICP, 20% intravenous mannitol 8 hourly was started and continued for 3 days. Diplopia disappeared on the 5th day with complete remission of LR paralysis, and the patient was advised discharge on the 6th postadmission day.


  Discussion Top


Dengue viral infection in the present case scenario presented with fever with chills and rigors for a brief period of 10 days. When symptoms worsened with severe bouts of vomiting, diplopia, and bleeding PV that developed acutely for 3 days, the patient reported immediately to the department of emergency medicine. Restricted ocular motility on dextroversion suggested right LR paralysis, however the patient described inconsistent diplopia and diplopia charting was unreliable, possibly because the patient was uncooperative and disoriented [Figure 3].
Figure 3: Globes in the left field of gaze

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Laboratory investigations showed reduced Hb concentration, PCV due to bleeding PV, and dehydration with elevated ESR that suggested an acute inflammatory response. Significant thrombocytopenia with a positive ELISA test for dengue NS1 Ag confirmed the presence of dengue viral infection. However, negative results were found for dengue IgM and other infectious diseases.

Hyponatremia (<130 mEq/l) could possibly explain the jeopardized nerve conduction process, concordantly confounded by the presence of early papilledema possibly to an actual rise of ICP manifesting as abducens nerve paralysis as a false localizing sign.[9] The pathophysiology of raising ICP in DF is still unclear and may be explored by conducting randomized clinical trials. We did not conduct nerve conduction studies, as the patient showed significant improvement in the general condition by conservative management. The pathogenesis of abducens nerve paralysis is debatable in this regard due to the imposition of dual mechanisms of raised ICP and hyponatremia.

Previously, subconjunctival and retinal hemorrhages showed spontaneous clinical resolution in 134 patients during an epidemic and concluded that hemorrhagic events occur with platelet counts <50,000/μL that was not observed in our study despite marked thrombocytopenia. Recently, fundal abnormalities have been described in Grade IV thrombocytopenia.[1],[10] The first case of LR paralysis was reported in a 40-year-old male from Malaysia, which was subsequently reported in 52- and 29-year-old male patients, in contrast to a 17-year-old young female patient in our study.[2],[3],[5] Previous studies concluded the possible pathogenic mechanisms as immune-mediated, demyelinating type of conduction defects, inflammatory vasculitis, multifactorial, and neurotrophic effect of dengue virus that play an important role [Table 1].[11] Notably, dengue-induced optic neuropathy occurred in three patients with spontaneous resolution in visual prognosis, and another study reported ocular flutter and truncal ataxia in a child.[7],[12]
Table 1: Previously published studies with year of publication and authors

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  Conclusion Top


The course of dengue-induced LR paralysis was transient and the pathogenesis was possibly raised ICP and/or hyponatremia that further merit research, which justifies this novel pathogenic mechanism. We recommend strong clinical vigilance of dengue viral infection in the presence of binocular diplopia, asymmetrical early papilledema, and electrolyte imbalance in acute febrile conditions with dehydration. Rapid clinical resolution of paralytic phase probably discriminates late remissions from other differential diagnoses of diplopia.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient and her guardian have given consent for the patient's images and other clinical information to be reported in the journal. The patient understands that her name and initial will not be published, and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Acknowledgment

We are grateful to the Head of Institution and Ophthalmology Department and specially thank the patient and her parents for their continuous and uninterrupted cooperation.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Kapoor HK, Bhai S, John M, Xavier J. Ocular manifestations of dengue fever in an East Indian epidemic. Can J Ophthalmol 2006;41:741-6.  Back to cited text no. 1
    
2.
Mishra A, Shukla S, Aggarwal S, Chaudhary B. Lateral rectus palsy in a case of dengue fever. Med J Armed Forces India 2015;71:S101-3.  Back to cited text no. 2
    
3.
Mazliha M, Boo YL, Chin PW. Isolated unilateral sixth cranial nerve palsy: A rare presentation of dengue fever. Malays Fam Physician 2016;11:25-6.  Back to cited text no. 3
    
4.
Carod-Artal FJ, Wichmann O, Farrar J, Gascón J. Neurological complications of dengue virus infection. Lancet Neurol 2013;12:906-19.  Back to cited text no. 4
    
5.
Shivanthan MC, Ratnayake EC, Wijesiriwardena BC, Somaratna KC, Gamagedara LK. Paralytic squint due to abducens nerve palsy: A rare consequence of dengue fever. BMC Infect Dis 2012;12:156.  Back to cited text no. 5
    
6.
Li GH, Ning ZJ, Liu YM, Li XH. Neurological manifestations of dengue infection. Front Cell Infect Microbiol 2017;7:449.  Back to cited text no. 6
    
7.
Sanjay S, Wagle AM, Au Eong KG. Optic neuropathy associated with dengue fever. Eye (Lond) 2008;22:722-4.  Back to cited text no. 7
    
8.
Donnio A, Béral L, Olindo S, Cabie A, Merle H. Dengue, a new etiology in oculomotor paralysis. Can J Ophthalmol 2010;45:183-4.  Back to cited text no. 8
    
9.
Vandergheynst F, Gombeir Y, Bellante F, Perrotta G, Remiche G, Mélot C, et al. Impact of hyponatremia on nerve conduction and muscle strength. Eur J Clin Invest 2016;46:328-33.  Back to cited text no. 9
    
10.
Malhotra R, Singh L, Bundela RK, Garg P, Kant D, Garg A, et al. Retinal profile: A clinical indicator of severity in dengue fever in a suburban Indian environment. Trop Doct 2014;44:143-7.  Back to cited text no. 10
    
11.
Murthy JM. Neurological complication of dengue infection. Neurol India 2010;58:581-4.  Back to cited text no. 11
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12.
Mahale RR, Mehta A, Buddaraju K, Srinivasa R. Parainfectious ocular flutter and truncal ataxia in association with dengue fever. J Pediatr Neurosci 2017;12:91-2.  Back to cited text no. 12
[PUBMED]  [Full text]  


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