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Paroxysmal autonomic instability with dystonia after pneumococcal meningoencephalitis.

Safadieh L, Sharara-Chami R, Dabbagh O - Case Rep Med (2012)

Bottom Line: The diagnosis of paroxysmal autonomic instability with dystonia was established.The patient responded to clonidine, baclofen, and benzodiazepines.Although this entity has been reported in association with traumatic brain injury, and as a sequel to some nervous system infections, this is the first case, to our knowledge, associated with pneumococcal meningoencephalitis.

View Article: PubMed Central - PubMed

Affiliation: Division of Neurology, Department of Pediatrics and Adolescent Medicine, American University of Beirut Medical Center, P.O. Box 11-0236, Riad El Solh, Beirut 1107 2020, Lebanon.

ABSTRACT
Streptococcus pneumoniae is a common cause of bacterial meningitis, frequently resulting in severe neurological impairment. A seven-month-old child presenting with Streptococcus pneumoniae meningoencephalitis developed right basal ganglia and hypothalamic infarctions. Daily episodes of agitation, hypertension, tachycardia, diaphoresis, hyperthermia, and decerebrate posturing were observed. The diagnosis of paroxysmal autonomic instability with dystonia was established. The patient responded to clonidine, baclofen, and benzodiazepines. Although this entity has been reported in association with traumatic brain injury, and as a sequel to some nervous system infections, this is the first case, to our knowledge, associated with pneumococcal meningoencephalitis.

No MeSH data available.


Related in: MedlinePlus

(a), (b) Brain magnetic resonance imaging one day after admission. (a) Axial apparent diffusion coefficient (ADC) map shows large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis. (b) Fluid attenuated inversion recovery (FLAIR) sequence showing normal brain tissue.
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fig1: (a), (b) Brain magnetic resonance imaging one day after admission. (a) Axial apparent diffusion coefficient (ADC) map shows large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis. (b) Fluid attenuated inversion recovery (FLAIR) sequence showing normal brain tissue.

Mentions: One day later, the child developed near continuous choreiform movements of upper extremities and bicycling of lower limbs alternating with dystonia of all limbs that spontaneously revolved during sleep. A brain magnetic resonance imaging, angiography, and venography study (MRI, MRA, MRV) revealed large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis, with no definite evidence for arterial or venous thrombosis (Figure 1(a)). No apparent lesions were demonstrated on routine MRI T1, T2, or fluid attenuated inversion recovery (FLAIR) sequences (Figure 1(b)). A 24-hour bedside video EEG study showed bilateral background slowing intermixed with sharp waves and sharply contoured theta activity. Several episodes of right-sided dystonic posturing were captured without any associated electroencephalographic correlates. Subsequently, phenytoin was discontinued and phenobarbital was added to valproic acid. Good therapeutic levels (phenobarbital level: 20–30 mg/L, valproic acid level: 50–70 mg/L) were maintained throughout his hospitalization.


Paroxysmal autonomic instability with dystonia after pneumococcal meningoencephalitis.

Safadieh L, Sharara-Chami R, Dabbagh O - Case Rep Med (2012)

(a), (b) Brain magnetic resonance imaging one day after admission. (a) Axial apparent diffusion coefficient (ADC) map shows large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis. (b) Fluid attenuated inversion recovery (FLAIR) sequence showing normal brain tissue.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3475321&req=5

fig1: (a), (b) Brain magnetic resonance imaging one day after admission. (a) Axial apparent diffusion coefficient (ADC) map shows large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis. (b) Fluid attenuated inversion recovery (FLAIR) sequence showing normal brain tissue.
Mentions: One day later, the child developed near continuous choreiform movements of upper extremities and bicycling of lower limbs alternating with dystonia of all limbs that spontaneously revolved during sleep. A brain magnetic resonance imaging, angiography, and venography study (MRI, MRA, MRV) revealed large areas of cortical and subcortical restricted diffusion in the frontal areas bilaterally, left parietal and left temporal regions, suggestive of encephalitis, with no definite evidence for arterial or venous thrombosis (Figure 1(a)). No apparent lesions were demonstrated on routine MRI T1, T2, or fluid attenuated inversion recovery (FLAIR) sequences (Figure 1(b)). A 24-hour bedside video EEG study showed bilateral background slowing intermixed with sharp waves and sharply contoured theta activity. Several episodes of right-sided dystonic posturing were captured without any associated electroencephalographic correlates. Subsequently, phenytoin was discontinued and phenobarbital was added to valproic acid. Good therapeutic levels (phenobarbital level: 20–30 mg/L, valproic acid level: 50–70 mg/L) were maintained throughout his hospitalization.

Bottom Line: The diagnosis of paroxysmal autonomic instability with dystonia was established.The patient responded to clonidine, baclofen, and benzodiazepines.Although this entity has been reported in association with traumatic brain injury, and as a sequel to some nervous system infections, this is the first case, to our knowledge, associated with pneumococcal meningoencephalitis.

View Article: PubMed Central - PubMed

Affiliation: Division of Neurology, Department of Pediatrics and Adolescent Medicine, American University of Beirut Medical Center, P.O. Box 11-0236, Riad El Solh, Beirut 1107 2020, Lebanon.

ABSTRACT
Streptococcus pneumoniae is a common cause of bacterial meningitis, frequently resulting in severe neurological impairment. A seven-month-old child presenting with Streptococcus pneumoniae meningoencephalitis developed right basal ganglia and hypothalamic infarctions. Daily episodes of agitation, hypertension, tachycardia, diaphoresis, hyperthermia, and decerebrate posturing were observed. The diagnosis of paroxysmal autonomic instability with dystonia was established. The patient responded to clonidine, baclofen, and benzodiazepines. Although this entity has been reported in association with traumatic brain injury, and as a sequel to some nervous system infections, this is the first case, to our knowledge, associated with pneumococcal meningoencephalitis.

No MeSH data available.


Related in: MedlinePlus