Clinical Characteristics and Functional Motor Outcomes of Enterovirus 71 Neurological Disease in Children

JAMA Neurol. 2016 Mar;73(3):300-7. doi: 10.1001/jamaneurol.2015.4388.

Abstract

Importance: Enterovirus 71 (EV71) causes a spectrum of neurological complications with significant morbidity and mortality. Further understanding of the characteristics of EV71-related neurological disease, factors related to outcome, and potential responsiveness to treatments is important in developing therapeutic guidelines.

Objective: To further characterize EV71-related neurological disease and neurological outcome in children.

Design, setting, and participants: Prospective 2-hospital (The Sydney Children's Hospitals Network) inpatient study of 61 children with enterovirus-related neurological disease during a 2013 outbreak of EV71 in Sydney, Australia. The dates of our analysis were January 1, to June 30, 2013.

Main outcomes and measures: Clinical, neuroimaging, laboratory, and pathological characteristics, together with treatment administered and functional motor outcomes, were assessed.

Results: Among 61 patients, there were 4 precipitous deaths (7%), despite resuscitation at presentation. Among 57 surviving patients, the age range was 0.3 to 5.2 years (median age, 1.5 years), and 36 (63%) were male. Fever (100% [57 of 57]), myoclonic jerks (86% [49 of 57]), ataxia (54% [29 of 54]), and vomiting (54% [29 of 54]) were common initial clinical manifestations. In 57 surviving patients, EV71 neurological disease included encephalomyelitis in 23 (40%), brainstem encephalitis in 20 (35%), encephalitis in 6 (11%), acute flaccid paralysis in 4 (7%), and autonomic dysregulation with pulmonary edema in 4 (7%). Enterovirus RNA was more commonly identified in feces (42 of 44 [95%]), rectal swabs (35 of 37 [95%]), and throat swabs (33 of 39 [85%]) rather than in cerebrospinal fluid (10 of 41 [24%]). Magnetic resonance imaging revealed characteristic increased T2-weighted signal in the dorsal pons and spinal cord. All 4 patients with pulmonary edema (severe disease) demonstrated dorsal brainstem restricted diffusion (odds ratio, 2; 95% CI, 1-4; P = .001). Brainstem or motor dysfunction had resolved in 44 of 57 (77%) at 2 months and in 51 of 57 (90%) at 12 months. Focal paresis was evident in 23 of 57 (40%) at presentation and was the most common persisting clinical and functional problem at 12 months (observed in 5 of 6 patients), with 1 patient also requiring invasive ventilation. Patients initially seen with acute flaccid paralysis or pulmonary edema had significantly greater frequencies of motor dysfunction at follow-up compared with patients initially seen with other syndromes (odds ratio, 15; 95% CI, 3-79; P < .001).

Conclusions and relevance: Enterovirus 71 may cause serious neurological disease in young patients. The distinct clinicoradiological syndromes, predominantly within the spinal cord and brainstem, enable rapid recognition within evolving outbreaks. Long-term functional neurological morbidity is associated with paresis linked to involvement of gray matter in the brainstem or spinal cord.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autonomic Nervous System Diseases / diagnosis
  • Autonomic Nervous System Diseases / epidemiology
  • Autonomic Nervous System Diseases / etiology*
  • Central Nervous System Viral Diseases / diagnosis
  • Central Nervous System Viral Diseases / epidemiology
  • Central Nervous System Viral Diseases / etiology*
  • Child, Preschool
  • Encephalitis, Viral / diagnosis
  • Encephalitis, Viral / epidemiology
  • Encephalitis, Viral / etiology
  • Encephalomyelitis / diagnosis
  • Encephalomyelitis / epidemiology
  • Encephalomyelitis / etiology
  • Enterovirus A, Human / isolation & purification
  • Enterovirus A, Human / pathogenicity*
  • Enterovirus Infections / complications*
  • Enterovirus Infections / diagnosis
  • Enterovirus Infections / epidemiology
  • Female
  • Follow-Up Studies
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • New South Wales / epidemiology
  • Paralysis / diagnosis
  • Paralysis / epidemiology
  • Paralysis / etiology