Abstract Chikungunya is usually a benign disease, and little is known on the occurrence of severe clinical complications. We describe a 12-year-old boy with rapid onset septic shock and multi-organ failure associated with chikungunya fever. Severe sepsis and septic shock can be associated with chikungunya. septic shock, multi-organ failure, chikungunya fever INTRODUCTION The chikungunya virus is an arthropod-borne virus transmitted by Aedes mosquitoes. In the past decade, chikungunya fever unexpectedly reemerged in the form of epidemics in many tropical African and Asian countries. In fact, it is now becoming a looming global threat . The chikungunya outbreak in Delhi in year 2016 was the worst in the past 6 years . Septic shock attributed to chikungunya infection is rare with only few cases reported in adult literature [3, 4]. We report a case of severe chikungunya sepsis, shock and multi-organ failure in an adolescent. CASE REPORT A 12-year-old male child presented with high-grade continuous fever, abdominal pain and pain in multiple small and medium joints for past 3 days, and watery loose stools and—five to six episodes of bilious vomiting for past 2 days. On arrival, he was irritable and had signs of hypotensive shock: heart rate 160/min, weak peripheral pulses, cold peripheries, capillary refill time >5 s and blood pressure 70/30 mm Hg. Systemic examination revealed distended, tense and tender abdomen. The rest of examination was normal. He received normal saline boluses and inotropic support (dopamine at 10 mcg/kg/min and epinephrine at 0.1 mcg/kg/min). Injections of ceftriaxone and teicoplanin were started. Central venous pressure and arterial pressure were 13 and 83/54 mm Hg, respectively. Venous blood gas (VBG) revealed pH 7.16, pCO2 43 mm Hg, pO2 54 mm, HCO3 15.3 mmol/l, lactate 4 mmol/l and central venous saturation (ScvO2) 77%. Investigations revealed hemoglobin 12.1 g percentage, total leukocyte count (TLC) 36 700/mm3 with neutrophil 80%, lymphocytes 3%, monocytes 1%, eosinophil 7% and platelet count 112× 103/mm3. Laboratory parameter revealed blood urea 95 mg/dl, serum creatinine 2.1 mg/dl, prothrombin time 19.5 s, international normalized ratio (INR) 1.51, activated partial thromboplastin time 36.7 s, serum sodium Na+ 132meq/l, potassium K+ 3.6meq/l, calcium Ca2+ 7.1meq/l, C-reactive protein 21.6 mg/l and serum albumin 2.3 g/dl. Abdominal radiograph and ultrasonography were suggestive of ileus with free fluid in abdomen. Initial blood culture was sterile. Dengue Ns1 antigen, IgM and IgG antibodies (by enzyme immunoassay), leptospira IgM & IgG antibodies (by immuno-chromatographic assay) and Weil Felix serology (by latex agglutination) were negative. Child continued to remain hemodynamically labile and required escalation of inotropic agents (dopamine at 10 mcg/kg/min, epinephrine at 0.2 mcg/kg/min and norepinephrine 0.3 mcg/kg/min). At 12 h, VBG showed worsening lactate to 11 mmol/l. Echocardiography revealed global left ventricular (LV) hypokinesia, LV systolic dysfunction with ejection fraction 35–40% and distended inferior vena cava. Epinephrine infusion was increased to 0.4 mcg/kg/min, and norepinephrine was decreased to 0.1 mcg/kg/min. Injection hydrocortisone was added and mechanical ventilation was initiated. However, child continued to deteriorate with labile hemodynamic status, falling urine output (from 0.8 to 0.2 ml/kg/h) and worsening organ dysfunctions. Slow low efficiency hemodialysis (SLED) started. Laboratory parameters showed lactate 4 mmol/l, ScvO2 54%, TLC 45 600/dl, N 82%, platelet count 23× 103/mm3, INR 1.81, urea 120 mg/dl and creatinine 1.8 mg/dl. At 36 h, he became severely hypotensive (BP 62/32 mm Hg). Normal saline and 5% albumin boluses of 30 ml/kg were given for stabilization. Antibiotics were upgraded to meropenem and clindamycin. Intravenous immunoglobulin, IVIG (2 g/kg) was given. Repeat cultures from blood, blind bronchial sample, urine and ascitic fluid were sent, and these cultures were sterile. He had two episodes of generalized seizures managed with levetiractetam. At 54 h, the child’s right pupil dilated and left pupil was mid-dilated with absent reaction to light bilaterally. Aggressive supportive care along with 3%normal saline continued. Non-contrast computed tomography brain showed small petechial bleed in left frontal and small hypodensity in left temporal and right frontal region. Subsequently fever improved, inotropic support decreased and SLED was continued. Chikungunya RNA polymerase chain reaction showed positive result. On Day 5, bilious aspirates and abdominal distension increased causing increased need of ventilator support. In addition, urine output was nil for 5 consecutive hours. Possibility of abdominal compartment syndrome and nosocomial infection was considered. Right flank abdominal drain was placed and ∼600 ml serous ascitic fluid drained. Gradually urine output, renal function and neurological function improved. He was extubated on Day 6. Blood culture from central line revealed growth of pseudomonas aeruginosa sensitive only to colistin. Central line was removed and injection colistin was added. Magnetic resonance imaging (MRI) brain revealed flair hyperintensity along bilateral high parietal, frontal lobes, centrum semiovale regions and corpus callosum. In addition, SWAN image revealed multiple blooming foci in corpus callosum and bilateral basal ganglia (Fig. 1). Child was discharged on Day 17 with final diagnosis of severe chikungunya, septic shock and multi-organ failure: encephalitis, myocarditis, acute kidney injury, disseminated intravascular coagulation and abdominal compartment syndrome. Child is on regular follow-up since past 7 months, and his neurological recovery is excellent. Fig. 1. View largeDownload slide (A and B) MRI Brain FLAIR axial images showing hyperintensity in subcortical white matter of frontal, parietal lobes and corpus callosum. (C) Axial SWAN image showing multiple blooming foci in corpus callosum and bilateral basal ganglia. Fig. 1. View largeDownload slide (A and B) MRI Brain FLAIR axial images showing hyperintensity in subcortical white matter of frontal, parietal lobes and corpus callosum. (C) Axial SWAN image showing multiple blooming foci in corpus callosum and bilateral basal ganglia. DISCUSSION The classical clinical manifestations of chikungunya are fever, rash and joint pain. In children, features are distinct from adults like less frequent rheumatological manifestations and more prevalent dermatological and hemorrhagic manifestations. The most characteristic features of the infection in infants were acrocyanosis and symmetrical superficial vesicobullous lesions. Though rare, reported neurological manifestations are febrile convulsions, focal seizures, encephalopathy, encephalitis, Guillain–Barre, encephalomyeloradiculitis and blindness because of retrobulbar neuritis. Other reported atypical clinical features are conjunctivitis, neuroretinitis, iridocyclitis, myocarditis, pericarditis, pneumonia, dry cough, nephritis, hepatitis, lymphadenopathy and pancreatitis . Chikungunya is usually a nonfatal disease; however, severe life-threatening complications can develop during acute phase. The reported case-fatality ratio is 1 in 1000 . Only recently, the two adult series have attributed severe sepsis and septic shock to chikungunya infection. In addition, the mortality rate of chikungunya septic shock was high (50–75%) in these two series [3, 4]. In present case, the differentials were bacterial septic shock and staphylococcal toxic shock syndrome; however, we did not isolate any other organism as a potential cause of septic shock except chikungunya virus. This finding suggests that chikungunya infection can rarely cause severe sepsis and septic shock in children. Present case is example of increasing documentation of viral septic shock with multi-organ failure. In our experience of chikungunya epidemic in year 2016, we have seen few children presenting with septic shock requiring inotropic support; however, multi-organ failure is rare. Although we have used IVIG in view of possible refractory toxic shock syndrome in present case, its role in chikungunya shock needs further research. In conclusion, chikungunya virus can lead severe septic shock and multi-organ failure in children. References 1 Thiberville SD, Moyen N, Dupuis-Maguiraga L, et al. Chikungunya fever: epidemiology, clinical syndrome, pathogenesis and therapy. Antiviral Res 2013; 99: 345– 70. Google Scholar CrossRef Search ADS PubMed 2 Chikungunya Outbreak in Delhi Worst in Last Six Years: Officials. Press Trust of India, Delhi. http://www.ndtv.com/delhi-news/chikungunya-outbreak-in-delhi-worst-in-last-six-years-officials-1460234 (14 April 2017, date last accessed). 3 Rollé A, Schepers K, Cassadou S, et al. Severe sepsis and septic shock associated with chikungunya virus infection, Guadeloupe, 2014. Emerg Infect Dis 2016; 22: 891– 4. Google Scholar CrossRef Search ADS PubMed 4 Torres JR, Leopoldo Códova G, Castro JS, et al. Chikungunya fever: atypical and lethal cases in the Western Hemisphere: a Venezuelan experience. IDCases 2014; 2: 6– 10. Google Scholar CrossRef Search ADS PubMed © The Author(s) . Published by Oxford University Press. All rights reserved. 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Journal of Tropical Pediatrics – Oxford University Press
Published: Jan 6, 2018
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