• Users Online: 110
  • Print this page
  • Email this page


 
 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 198-200

Cerebral visual impairment secondary to chikungunya encephalitis – A case report


1 Department of Pediatric Ophthalmology and Strabismus, Aravind Eye Hospital, Coimbatore, Tamil Nadu, India
2 Consultant Radiologist , Columbia Asia Hospital, Hebbal, Bengaluru, Karnataka, India
3 Resident in Ophthalmology , MD, MUSC, South Carolina, USA
4 Private Practice Ophthalmologist, Salina, Kansas, USA

Date of Submission03-Apr-2021
Date of Acceptance12-Jul-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Sandra C Ganesh
Department of Pediatric Ophthalmology and Strabismus, C.2, Staff Quarters, Aravind Eye Hospital, Avinashi Road, Coimbatore - 641 014, Tamil Nadu
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_754_21

Rights and Permissions
  Abstract 


Cerebral visual impairment is a significant cause of visual impairment in children in developing countries. It has varied etiologies. In this report, we describe the evaluation and management of a 1-year-old child with visual impairment due to neonatal chikungunya encephalitis.

Keywords: Cerebral visual impairment, chikungunya encephalitis, vision training


How to cite this article:
Ganesh SC, Shailaja C, Narayanasamy V, Sagar G D, Sindhu A, Rao SG, House R, Lawrence L. Cerebral visual impairment secondary to chikungunya encephalitis – A case report. Indian J Ophthalmol Case Rep 2022;2:198-200

How to cite this URL:
Ganesh SC, Shailaja C, Narayanasamy V, Sagar G D, Sindhu A, Rao SG, House R, Lawrence L. Cerebral visual impairment secondary to chikungunya encephalitis – A case report. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Aug 14];2:198-200. Available from: https://www.ijoreports.in/text.asp?2022/2/1/198/334976



Cerebral visual impairment (CVI) is defined as visual impairment due to lesions of the retrogeniculate visual pathways not explained by ocular pathology.[1] CVI has emerged as a significant cause of profound visual impairment in children. The common causes of CVI include periventricular leukomalacia, perinatal hypoxia–anoxia, hypoglycemia, central nervous system infections, hydrocephalus, epilepsy, trauma, and congenital malformations.[2],[3] Viral meningoencephalitis in the early neonatal period is a known cause of CVI.[4] Here, we report a rare case of CVI secondary to chikungunya viral encephalitis in an infant.


  Case Report Top


A 1-year-old male child was brought to our pediatric ophthalmology clinic in a large outpatient eye care center in south India by his parents after noticing visual inattention. He had a history of viral encephalitis in the early neonatal period and was hospitalized on the sixth day of life due to fever, jaundice, and poor feeding. The investigations revealed thrombocytopenia (platelet count – 35,000/mcL) and elevated C reactive protein (13.70 mg/L). Serology was positive for chikungunya immunoglobulin M antibodies (rapid antibody test kit). Magnetic resonance imaging (MRI) of the brain with diffusion-weighted imaging at birth [Figure 1] showed areas of restricted diffusion in the corpus callosum, bilateral thalamus, and frontotemporal and parietal subcortex, as well as periventricular white-matter lesions suggestive of viral encephalitis. He also had hyperpigmentation of the skin with a macular rash during the recovery period.
Figure 1: MRI (magnetic resonance imaging) taken on the seventh day of life. (a) Axial DWI (diffusion-weighted imaging) demonstrating restricted diffusion in corpus callosum (blue arrows), bilateral frontoparietal white matter (green arrows), and bilateral thalami (yellow arrow). (b) Axial DWI image demonstrating extensive restricted diffusion in bilateral frontoparietal white matter (yellow arrows)

Click here to view


The developmental assessment was positive for delay in gross and fine motor skills with rocking behavior. Functional vision assessment with LEA GRATINGS showed 1.0 cycles per cm (57 cm) and fixation only on high-contrast control of Hiding Heidi low-contrast face test. There was inconsistent fixation and significant delay in visual response with a down gaze preference. Motility exam demonstrated a left esotropia, and cycloplegic retinoscopy was age-appropriate. Anterior segment evaluation including pupillary reaction was normal. Fundus evaluation revealed bilateral temporal optic disc pallor. A repeat MRI brain at 1 year of age [Figure 2], [Figure 3], [Figure 4] showed thin corpus callosum, significant atrophy of frontal parenchyma, and hypomyelination changes consistent with postencephalitic sequelae. CVI was diagnosed, and appropriate developmental interventions were demonstrated to the parents by the early intervention team. Examples of activities included play with an illuminated object in a dark room, use of high-contrast toys to gain visual attention, and mirror play to integrate training with play to achieve better participation.
Figure 2: Follow-up MRI (magnetic resonance imaging) taken at 1 year of age. Axial FLAIR (fluid-attenuated inversion recovery) image demonstrating gliotic changes with volume loss of bilateral frontal lobes. Gross thinning of the genu of corpus callosum noted (blue arrow)

Click here to view
Figure 3: Coronal T2-weighted image demonstrating bilateral frontal lobe atrophy with widened interhemispheric fissure (yellow arrow) and sulci. Prominent extra-axial CSF (cerebrospinal fluid) spaces noted over bilateral frontal lobes (green arrows). The above features indicate bilateral frontal lobe atrophic changes

Click here to view
Figure 4: Sagittal T1-weighted image demonstrating gross diffuse atrophy of corpus callosum (yellow arrows)

Click here to view


Two months after the initial visit, a live internet-based teleconsultation was arranged, which included an international early intervention expert, the involved pediatric ophthalmologist, and the child's occupational therapist. Videos demonstrating visual function were sent in advance to the intervention team for review. The live, online session allowed the child to be assessed in a comfortable and familiar home environment. Additional interventional strategies suggested by the team included tactile information to supplement visual input, involvement of the patient's older sibling in play, improved contrast in the home, and a clutter-free environment. The child's local interventionist was given feedback on best practices to train the family and help with the neurodevelopmental outcomes of the child.

After a total of 3 months of therapy, the child was reexamined in the clinical setting. Parents reported improved responses to familiar objects and more rapid visual responses in the home environment. On examination, the child was able to fix and follow objects, although visually guided reach was still absent. LEA GRATINGS showed 2.0 cycles per cm (57 cm), and he was able to differentiate the 25% contrast card of Hiding Heidi. Delayed and inconsistent fixation remained. The family was advised on further interventions for vision as well as physiotherapy for his delay in motor function.


  Discussion Top


Chikungunya virus belongs to the family Togaviridae and genus Alphavirus. Human transmission is vector-borne through Aedes aegypti and Aedes albopictus mosquitoes. Vertical transmission from mother to child has also been reported.[5],[6] According to the most available evidence, transmission occurs during the intrapartum period[7] at a rate close to 50% for viremic women, and cesarean section does not appear to be protective in vertical transmission. In this case, there was no documented evidence of chikungunya infection in the mother; however, intrapartum transmission seems to be the most plausible.

Encephalopathy is the most common complication among neonates infected by chikungunya. A study from Reunion island described 739 mothers with chikungunya infection out of which 39 were symptomatic during the intrapartum period.[8] Nineteen infants developed acute disease within 3 to 7 days of delivery, with nine cases (47%) developing encephalopathy. Clinical features in affected neonates included fever, poor feeding, petechiae, and maculopapular rash. MRI demonstrated severe white-matter injury and demyelination changes. In addition to acute manifestations, there is evidence for impact on long-term development of these neonates. In a study investigating the neurodevelopmental outcomes in infants with perinatal mother-to-child chikungunya infection, Gérardin et al.[9] found that neurocognitive outcomes were worse in infected children compared with nonmatched controls. A history of encephalopathy from perinatal chikungunya infection led to the poorest neurocognitive outcomes.


  Conclusion Top


In conclusion, mothers with symptoms of fever, rash, and arthralgia during the perinatal period need serological testing in areas where the virus is endemic. Neonates with a suspected infection should be evaluated early for visual behavior along with management of the systemic disease. They need follow-up for at least 2 years for evidence of neurodevelopmental delay regardless of the initial presentation. The neurological impact of perinatal infection such as encephalopathy may not be obvious at birth; however, these children should be considered high risk for neurodevelopmental delay or disability, including visual impairment, and continual assessment and intervention strategies may be indicated. This rare case of neonatal chikungunya encephalitis leading to CVI demonstrates the need for early intervention and serial follow-up of exposed neonates for the neurodevelopmental delay and visual dysfunction.

Declaration of patient consent

A statement of consent to publish this case and images was gathered from the patient.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Boot FH, Pel JJ, van der Steen J, Evenhuis HM. Cerebral visual impairment: Which perceptive visual dysfunctions can be expected in children with brain damage? A systematic review. Res Dev Disabl 2010;31:1149-59.  Back to cited text no. 1
    
2.
Soul J, Matsuba C. Visual impairment in children due to damage to the brain. In: Dutton GN, Bax M, editors. Causes of Damage to the Visual Brain. London, England: Mac Keith Press; 2010. p. 20-6.  Back to cited text no. 2
    
3.
Chhablani PP. Cortical visual impairment. Sci J Med Vis Res Foun 2015;XXXIII: 148-9.  Back to cited text no. 3
    
4.
Gunawan PI, Primayani D, Saharso D. Cortical visual impairment in children with acute encephalitis syndrome. Bangladesh J Medical Sci 2016;15:249-51.  Back to cited text no. 4
    
5.
Torres JR, Falleiros-Arlant LH, Dueñas L. Congenital and perinatal complications of chikungunya fever. A Latin American experience Int J Infect Dis 2016;51:85-8.  Back to cited text no. 5
    
6.
Evans-Gilbert T. Chikungunya and neonatal immunity: Fatal vertically transmitted chikungunya infection. Am J Trop Med Hyg 2017;96:913-5.  Back to cited text no. 6
    
7.
Villamil-Gómez W, Alba-Silvera L, Menco-Ramos A, Gonzalez-Vergara A, Molinares-Palacios T, Barrios-Corrales M, et al. Congenital Chikungunya virus infection in Sincelejo, Colombia: A case series. J Trop Pediatr 2015;61:386-92.  Back to cited text no. 7
    
8.
Gérardin P, Barau G, Michault A, Bintner M, Randrianaivo H, Choker G, et al. Multidisciplinary prospective study of mother-to-child chikungunya virus infections on the island of La Réunion. PLoS Med 2008;5:e60.  Back to cited text no. 8
    
9.
Gérardin P, Sampériz S, Ramful D, Boumahni B, Bintner M, Alessandri J-L, et al. Neurocognitive outcome of children exposed to perinatal mother-to-child chikungunya virus infection: The CHIMERE cohort study on Reunion Island. PLoS Negl Trop Dis 2014;8:e2996.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed324    
    Printed4    
    Emailed0    
    PDF Downloaded38    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]