Acute flaccid paralysis and seizure induced pneumothorax can ...

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CASE REPORT

Acute flaccid paralysis and seizure induced pneumothorax can accompany Japanese encephalitis Sugata Dasgupta, Arunima Chaudhuri1, Pritam Biswas2, Saibal Si2

Abstract

Department of Anaesthesiology, Division of Critical Care Medicine, RG Kar Medical College, 2Department of Anaesthesiology and Critical Care, RG Kar Medical College, Kolkata, 1Department of Physiology, Burdwan Medical College, Burdwan, West Bengal, India Address for correspondence: Dr. Arunima Chaudhuri, Krishnasayar South, Borehat, Burdwan - 713 102, West Bengal, India. E-mail: [email protected]

A 14-year-old boy from Southern parts of West Bengal, India presented in a Tertiary Care Hospital of West Bengal with high-grade fever and acute flaccid paralysis, later proved to be a case of Japanese encephalitis. His clinical course was also complicated by an unexpected occurrence of seizure-induced pneumothorax. The patient received supportive therapy and ultimately discharged from hospital with residual flaccid paralysis of lower limbs as the only neurological impairment. We hereby report these two rare accompaniments of Japanese encephalitis, a common occurrence in Asian countries.

Key words: Acute flaccid paralysis, Japanese encephalitis, pneumothorax

INTRODUCTION Japanese encephalitis (JE) is a common mosquitoborne flavivirus related viral encephalitis and most common cause of vaccine preventable encephalitis syndrome in Asian countries.[1-3] It is transmi ed in zoonotic cycle among mosquitoes and vertebrate hosts, chiefly pigs, and wading birds. It is spread through these regions by culicine mosquitoes, most often Culex tritaeniorhynchus.[1-4] Humans act as dead-end host as they do not develop enough viremia to infect the mosquitoes. Less than 1% people infected with JE virus (JEV) develop clinical illness typically a er an incubation period of 5-15 days.[1-4] Among the different neurologic symptoms headache, convulsion (66% in children), meningismus (more common in adults), altered mental status, cranial nerve palsies (found in Access this article online

33% patients) generalized weakness, pathologic reflexes, and a syndrome of acute flaccid paralysis have been described.[1-7] We report a case of JE presenting with initial complaints of fever and ascending paralysis involving both limbs and right leg.

CASE REPORT A 14-year-old boy, resident of North-24-paraganas, West Bengal, India, presented with initial complaints of fever, acute onset ascending paralysis of 5 days duration. On initial clinical examination, all the jerks were normal except bilateral ankle jerk which were diminished and neck rigidity was present. The well alert and conscious child at the time of admission gradually developed worsening of sensorium to the extent that he needed to be intubated for airway protection in next 48 h. He further suffered from several episodes of generalized tonic-clonic seizures.

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DOI: 10.4103/0974-5009.175451

Blood counts, electrolytes were within normal limits, and serology screening (HIV I and II, HBsAg, anti-HCV Ab.), MPDA, dengue IgM, NS1 were negative. Cerebrospinal fluid (CSF) examination showed high protein (147 mg/dL), glucose (67 mg/dL), cell count of 330/cmm (88% neutrophil), adenosine deaminase was Journal of the Scientific Society, Vol 43 / Issue 1 / January-April 2016

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within normal limits and CSF aerobic and anaerobic culture were negative. The diagnosis of JE was based on the presence of anti-JE virus IgM in the CSF and seroconversion of IgM and IgG by the enzymelinked immunosorbant assay (ELISA) method. Nerve conduction study showed nonrecordable F responses from common peroneal nerves bilaterally, bilateral tibial nerves showed ill-persistently present ‘F’-response, all sensory nerve action potentials (SNAPs) were normal. Motor conduction studies of bilateral median, ulnar, tibial, and peroneal nerves were normal with normal distal latency, compound motor action potential (CMAP) amplitudes, and conduction velocities (CMAP; SNAP). Nerve conduction study interpretation was demyelinating polyradiculopathy (as F waves were absent). The unique feature of this LMN involvement in JE is the involvement of radicles. Involvement of anterior horn cell has been reported, but rediculopathy is quite an unusual finding. His clinical course was complicated with an unexpected occurrence of bilateral pneumothorax (due to seizure) in-spite of predicted body weight adjusted tidal volume limited controlled invasive ventilation and absence of any surgical procedures (no central line catheter [CVC] lines). Though X-ray couldn’t delineate the diagnosis, highresolution computed tomography (HRCT) lungs were confirmatory. Bilateral chest drains were placed for it. Tracheotomy was required a er 10 days of ventilation. The child gradually improved, had be er sensorium and orientation over next 10 days, gradually weaned off ventilator and decannulated. The boy had recovered his upper limb power, but legs continue to be flaccidly paralyzed till date.

DISCUSSION Acute flaccid paralysis as a presenting component of JE is rarely reported in the literature.[1,2] Therefore, the clinical scenario of this boy offers a differential diagnosis for cases where other more common differentials for acute flaccid paralysis have already been excluded. Seizure induced pneumothorax that too in an intubated and ventilated patient also is a rare event. The term occult pneumothorax describes a pneumothorax that is not suspected on the basis of clinical examination or plain radiography, but is ultimately detected with thoracoabdominal computed tomography (CT). [8,9] In the present case, HRCT confi rmed the diagnosis of pneumothorax though radiograph did not reveal the disorder. Seizure induced pneumothorax is also a rare complication. Review of literature shows pulmonary complications of seizures include the aspiration of gastric contents 28

and neurogenic pulmonary edema. The patient’s clinical course was complicated with an unexpected occurrence of bilateral pneumothorax in-spite of predicted body weight adjusted tidal volume limited controlled invasive ventilation and absence of any surgical procedures (no CVC lines). Hence, seizure-induced pneumothorax was suspected. Gastric inflation in intubated patients may interfere with eff ective ventilation and cause regurgitation, pneumothorax, muscle twitching, and seizures, especially in patients with poor cardiac output and hepatic or renal failure.[10] Japanese encephalitis has got significant mortality and morbidity risks and spreads in epidemics, but it is also a vaccine preventable and amenable to environmental measures.[1-7] Therefore, it will be prudent to keep this rare presentation also in mind to track those epidemics early. On the other hand, once infected it has got no specific therapy, therefore, rare events like pneumothorax needs to diagnosed and managed early to tide over the emergencies. Chung et al. in 2007[1] in Taiwan reported case of a young adult man who received four doses of JEV (Nakayama strain) vaccination in childhood, but still developed acute JE virus infection, characterized with acute flaccid paralysis. A er supportive care, he was weaned from the mechanical ventilator and at discharge 1-month later, his muscle power level and deep tendon reflexes recovered partially. The diagnosis of JE was based on the presence of anti-JE virus IgM in the CSF and seroconversion of IgM and IgG by the ELISA method. They concluded that Japanese virus encephalitis infection should be considered as a differential diagnosis in any adult presenting with acute flaccid paralysis. Hossain et al. in Bangladesh in 2005[2] reported a case of an 11-year-old boy with fever and unconsciousness. His pupillary light reflexes were bilaterally sluggish, all the deep tendon reflexes were diminished, and planter reflexes were bilaterally flexor. His left leg became paralyzed on 13th day and by 15th day all his limbs became paralyzed. Besides these, the patient had gross muscle wasting, but his deep tendon reflexes were diminished. The patient was proved to have JE infection by a four-fold rise in virus-specific antibody detected in paired acute and convalescent sera by ELISA. Solomon et al. in 2008[9] conducted a study to assess the field-test version of the new WHO JE surveillance standards. They applied the clinical case definition of the acute encephalitis syndrome (AES), laboratory diagnostic criteria and case classifications to patients

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with suspected central nervous system (CNS) infections in southern Viet Nam. Findings of the 380 patients (149 children) recruited with suspected CNS infections, 296 (96 children) met the AES case definition. 54 children were infected with JEV, of whom 35 (65%) had AES, giving a sensitivity of 65% (95% confidence interval [CI]: 56-73) and specificity of 39% (95% CI: 30-48). Nine adults with JEV presented with AES. 19 JEV-infected children missed by surveillance included 10 with acute flaccid paralysis, two with flaccid hemiparesis and six with meningism only. Altering the case definition to include limb paralysis and meningism improved sensitivity to 89% (95% CI: 83-95), while reducing specificity to 23% (95% CI: 15-30). Six children that did not have AES on admission had reduced consciousness after admission. CSF analysis diagnosed seven patients negative on serum analysis. Five patients with neurological manifestations of dengue infection had JEV antibodies in serum and would have been misdiagnosed had if not tested for dengue antibodies in parallel. Children infected with JEV that presented with acute limb paralysis or neck stiffness only were missed by the surveillance standards in the above study, although some of them subsequently became encephalopathic.

CONCLUSION Flaccid paralysis can be a presenting component of JE and seizure-induced pneumothorax can occur even in an intubated and ventilated patient.

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10. Kleinman ME, Chameides L, Schexnayder SM, Samson RA, Hazinski MF, Atkins DL, et al. Part 14: Pediatric advanced life support: 2010 American Heart Association Guidelines for Cardiopulmonary Resuscitation and Emergency Cardiovascular Care. Circulation 2010;122 18 Suppl 3:S876-908. How to cite this article: Dasgupta S, Chaudhuri A, Biswas P, Si S. Acute flaccid paralysis and seizure induced pneumothorax can accompany Japanese encephalitis. J Sci Soc 2016;43:27-9. Source of Support: Nil. Conflicts of Interest: None declared.

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