Perinatal chikungunya in twins.

We report a case of vertically transmitted chikungunya infection in heterozygous twin neonates presenting as seizures, encephalopathy, midfacial hyperpigmentation, anemia, and thrombocytopenia. This could be considered as a rare cause of neonatal seizure and identification would help in appropriate management.

Introduction

Chikungunya infection in the newborn period is a rare entity. It is due to an arbovirus which is commonly transmitted by mosquito bite. We describe a vertically transmitted infection in heterozygous twin neonates whose characteristic clinical picture clinched the diagnosis.

Case Report

Twin 1

Male baby, first born of dichorionic diamniotic twin pregnancy, at 36 weeks weighing 2528 g delivered by spontaneous vaginal delivery to a 27 years G3P2L2 mother, with normal perinatal transition, presented with multiple episodes of seizures on day 5 of life. Upon initial workup, the baby was found to have hypocalcemia which was corrected, but the seizures persisted, requiring intravenous phenobarbitone and phenytoin for control. The baby, then developed encephalopathy and persistent apnea, requiring mechanical ventilation for 4 days. On day 8, the baby was weaned off the ventilator by which time hyperpigmentation over midfacial area was noted. Magnetic resonance imaging (MRI) brain revealed hemorrhagic leukoencephalopathy [Figure 1]. Investigations showed total count – 18,600/m3, differential count – neutrophils 36%, lymphocytes – 52%, eosinophils – 2%, monocytes – 10%; hemoglobin – 8.3 g/dl, platelet – 25,000/mm3. There was mild uremia which resolved within 3 days. Cerebrospinal fluid and blood cultures were sterile. In view of seizures and encephalopathy associated with midfacial hyperpigmentation, anemia, thrombocytopenia, with maternal fever during delivery and similar picture in the second twin baby, a possibility of vertically transmitted chikungunya infection was considered. The baby's serum IgM for chikungunya was positive. The maternal serum IgM was also positive for chikungunya. The baby was treated with platelet transfusion, antibiotics and supportive measures. He improved, was put on direct breastfeeds and was discharged on day 24 of life.

Figure 1

(a) Magnetic resonance imaging showing hemorrhagic leukoencephalopathy (b) magnetic resonance imaging brain showing hemorrhagic leukoencephalopathy

Twin 2

Girl baby, second born of dichorionic diamniotic twin pregnancy, at 36 weeks weighing 2503 g delivered by spontaneous vaginal delivery to a 27 years G3P2L2 mother. The perinatal transition was normal. This baby also had multiple seizures on day 5 of life, with encephalopathy needing mechanical ventilation for 12 h after which she self-extubated. The baby was encephalopathic, had other features such as midfacial hyperpigmentation, anemia, and thrombocytopenia, which were identical to the other twin. MRI showed hemorrhagic leukoencephalopathy [Figure 1]. Serum IgM for chikungunya was positive. The baby's condition improved and was discharged on day 24 of life.

Discussion

Chikungunya virus (CHIKV) is an arthropod-borne, alphavirus belonging to the family Togaviridae. It is transmitted by the virus carrying female Aedes mosquito (Aedes albopictus and Aedes aegypti). It was first recognized as an epidemic in East Africa in 1952–1953.[1] In India, the virus was first isolated in Kolkata, in 1963,[2] and then there was an epidemic in 2005.[3] Vertically transmitted infection was first reported in La Reunion Island.[4] The word chikungunya means “to walk bent over” and refers to the effect of the excruciating joint pain that characterizes this infection. The diagnosis is by detection of IgM antibodies by ELISA or reverse transcription-polymerase chain reaction.[5] Prevention is by vector control strategies to protect against mosquito bites.

Perinatally transmitted CHIKV leads to encephalitis in the newborn.[6] The risk is greatest during birth, especially if the mother is affected days before delivery. Infection during pregnancy may lead to abortion, preterm labor. Mangalgi et al. studied 8 cases of vertically transmitted infection all of whom had perioral pigmentation and encephalopathy[7] The newborn presents on day 3–5 with fever, excess crying, nasal blotchy erythema, freckle-like pigmentation over midfacial area, maculopapular rash, even vesiculobullous lesions, shock, apnea, disseminated intravascular coagulation, and neurological manifestations such as seizures and altered sensorium. Neonatal encephalitis due to CHIKV can be associated with high mortality and morbidity. The neurodevelopmental outcome could be affected in the long run and more severe the encephalopathy, poorer the prognosis.[6] Pialoux et al. found that majority of neonatal chikungunya had severe manifestations.[8] Differential diagnosis includes septicemia, pyogenic meningitis, and metabolic encephalopathy. Treatment is purely symptomatic. Thrombocytopenia should be managed promptly to prevent intracerebral bleeds.[9] No specific antivirals are found to be useful and no vaccine has been invented as yet.

Conclusion

Pediatricians and neonatologists should have a high index of suspicion for perinatal or neonatal chikungunya in endemic areas in any newborn presenting with seizure, encephalopathy, and skin manifestations. A detailed maternal peripartum history is useful. MRI is necessary for prognostication and close follow-up for neurodevelopmental outcome is important.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.