Although there are few anecdotal reports on the vertical transmission of the virus
from the mother to the newborn [1-3], there are no reports on
follow up of these children. We describe the follow up of two
such newborns with encephalopathy at 3years.
Case Report
Case 1: A 5 day old male, born at term by
caesarian section with birth weight of 3.5 kg and normal apgar
scores was referred with altered sensorium and convulsive apnea.
On examination, he had features of encephalopathy. Mother had
history of fever with joint pain few days prior to delivery.
Initial work up for seizures was normal (blood glucose, serum
electrolytes, CSF examination and CT scan). Septicemia was ruled
out by relevant investigations (complete blood count, peripheral
smear, CSF examination and blood culture sensitivity). He had
hypoproteinemia and lymphedema during the hospital stay. At
1week of life, he was noticed to have hyperpigmentation over
nose, face and groin. In view of maternal fever and joint pain
and endemicity of the disease, baby’s and maternal blood was
sent for RT PCR for chikungunya, which was positive. Hence, a
final diagnosis of vertical transmission of chikungunya was
considered. The baby was treated symptomatically and was
discharged on direct breast feeding. The hyperpigmentation
lasted two months and gradually settled.
During his initial follow up, he was found to
have hypertonia for which he was advised early intervention and
stimulation. He subsequently developed spastic diplegia. He also
has a seizure disorder starting at 11months for which he is on 2
anticonvulsants, CT scan and EEG done at one and a half years
were normal. His IQ assessment done by the Binet Kamat test of
intelligence showed a score of 62 and his category of IQ was
borderline. His social intelligence was age appropriate. His
meaningful memory and visuo-motor skills were adequate whereas
his language, nonmeaningful memory, conceptual thinking,
nonverbal and numerical reasoning were inadequate. He was also
found to be hyperactive for which he is on appropriate
intervention.
Case 2: A 5-day-old female baby
born at term with birth weight of 2.8 kg to HBsAg positive
mother by caesarian section was referred with repeated
convulsive apnea and lethargy since 2 days. Mother had history
of high grade fever with joint pain just prior to delivery. On
admission the baby had features of encephalopathy. Initial
workup for seizures was normal (blood glucose and electrolytes,
CSF, CT scan and EEG). Work up for septicemia was also negative.
Baby was found to have hypoproteinemia and lymphedema. Baby had
to be ventilated on day 4 of admission in view of repeated
convulsive apnea and poor respiratory efforts. Supportive
treatment was given baby and was discharged on full feeds.
Babies and maternal serum for RT PCR for chikungunya positive.
On day 10, baby was noticed to have perioral, limb and abdominal
hyperpigmentation. On follow up, she was found to be hypotonic
for which she was started on early intervention and stimulation.
At 6-months of age, she was found to have poor visual regard.
VEP was done and was found to be abnormal with poor NPN
complexes suggestive of primary optic atrophy. BAER study done
at one year of age was normal. She is presently 3 yrs and has
hypotonic cerebral palsy with mental retardation. Her IQ
assessment done by Binet Kamat test of Intelligence gave a score
of 58 and the category of IQ was poor. Her social intelligence,
visuo-motor skills, numerical reasoning and language skills were
poor. Her conceptual thinking, non verbal reasoning and
meaningful memory were also inadequate whereas her non
meaningful memory was adequate.
Discussion
We found that both the newborns that
developed chikungunya encephalopathy had persistent disabilities
which included cerebral palsy, visual impairment, seizure
disorder and behavioral problems. Chikungunya virus infection
was first reported to affect the nervous system in the 1960s[4]. The neurotropism of this virus has
not been completely studied. The ability of the virus to invade
and replicate in the brain parenchyma has not been consistently
proven by animal studies. Experimental studies have shown that
the virus disseminates to the central nervous system in severe
cases, where it specifically targets the choroids plexus and the
leptomeninges [5]. In a study
done in adults in Nagpur district of Maharashtra,
it was found that 16.3% had neurological complications which
included encephalitis, myelopathy, peripheral neuropathy,
myeloneuropathy and myopathy. RT-PCR and real time PCR was
positive in the CSF in 16% and 18%, respectively [6]. Another
report [1]from the
Reunion Island showed that 4 out of the 9 neonates with
encephalopathy developed persistent disabilities, which included
cerebral palsy with blindness and ataxia in one and three had
ocular and behavioral or postural deficiencies. However, the
outcome of the neurological symptoms was generally good in
adults [7].
In conclusion, encephalopathy appears to be
the most common clinical presentation of the disease during
mother to child transmission and can be associated with long
term disability. Hence, newborns with vertical transmission of
chikungunya need a close follow up for abnormal
neurodevelopmental outcome.
Contributors: Both authors contributed to
review of literature and drafting the manuscript.
Funding: None; Competing interests:
None stated.
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