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Neurology Case of the Week
HASSANAIN TOMA, MD
NEUROLOGY PGY-4
M O V E M B E R 2 ND, 2 0 1 2
Chief Complaint
Altered mental status:
Lethargic -> obtunded
HPI
5 yo Caucasian boy
Admission - 11 days:
Fever of 104 -> diagnosed with a sore throat and placed on antibiotics
Admission - 8 days:
Diarrhea after -> stopped abx -> diarrhea continued.
coughing, nasal congestion, and rhinorrhea
Admission - 7 days
Vomiting x 4 days, decreased PO intake.
Admission -1 day
crying episodes.
Admission:
Abdominal pain, screaming due to pain
He was transferred here for further evaluation his abdominal pain.
Next day became increasingly lethargic, and was intubated for airway protection.
GROWTH/DEVELOPMENT:
Growth delay and mild
development delay.
Able to walk, speaks.
Attends kindergarten.
PAST
MEDICAL/SURGICAL/BIRT
H HISTORY:
Eczema
Hypothyroidism
milk allergy (has since grown out
of this per mom)
ADHD.
Adverse Reactions/Allergies:
FAMILY HISTORY:
Asthma, HTN.
SOCIAL HISTORY:
PAST SURGICAL
Milk Products(Rashes)
No surgeries.
Parents live separately.
Preston spends time at each
parent's house.
Dad smokes outside the home.
MEDICATIONS:
Levothyroxine
Antacid
strattera
IMMUNIZATIONS:
Up to date per mom.
Physical Exam
General: Intubated, appears of stated age. No
spontaneous movement.
Head/Neck: Microcephalic. No neck masses.
Eyes: PERRL. Erythema of conjunctiva of left
eye.
ENT: TM's pearly and nonbulging bilaterally. No
erythema or exudate of oropharynx. Dry lips.
Mental State: Obtunded, not responsive to
stimuli.
CN II: PERRL slow reacting.
CN III & IV: Positve dolls.
CN V: Grimaces pain. Corneal reflex preserved
in both eyes.
CN VI: Unable to access extra ocular
movements intact bilaterally.
CN VII: Symmetrical face.
CN VIII: Unable to assess hearing.
CN IX & X: Gag present.
Chest: CTAB, no wheezing.
CV: RRR, no murmurs, rubs, or gallops.
Abdomen: abdomen is soft. Non distended.
+BS
Lymph: No cervical LAD.
Skin: No rashes seen on visible skin.
Motor: The tone is hypertonic with rigidity.
Sensory: withdraws to pain.
Reflexes: 3 diffusely. Upgoing toes.
Coordination: could not be tested.
Gait: could not be tested.
Labs
HEMATOLOGY
WBC
HGB
HCT
35.2%
Platelet
182
% Band
32.9 %
9.46
12.4
URINALYSIS/FECES
Color Ur
STRAW
Clarity Ur
CLEAR
Specific Gravity Ur
1.030
pH Ur
6.0
Glucose Ur
NEGATIVE
Ketones Ur
2+
A
Protein Ur
NEGATIVE
Blood Ur
NEGATIVE
Bili Ur
NEGATIVE
Urobilinogen Ur
NORMAL
Nitrite Ur
NEGATIVE
Leukocytes Ur
NEGATIVE
WBC Ur
1-4
RBC Ur
1-4
Bacteria Ur
NONE
Renal Epithelial Cells Ur FEW
Casts Ur
NONE
Crystals Ur
NONE
CHEMISTRY
Sodium
Potassium
Chloride
Carbon Dioxide
Anion Gap
Calcium
Glucose
BUN
Creatinine
C Reactive Prot
Protein Total
Alb
Bili, Total
Bili, Direct
Bili, Indirect
AST
ALT
AP
Amylase
Lipase
Sed Rate
CSF
Clarity
Color
RBC
WBC
Glucose
Protein
137
4.0
101
22
14
9.3
93
10
0.31
2.3 H
6.5
3.6
0.6
0.0
0.3
146 H
138 H
143
70
308 H
34H
ENDOCRINOLOGY
TSH
3.02
T4 Free
1.5
CLEAR
COLORLESS
0
2
79
41
What??
Where??
Differential Diagnosis
MRI - DWI
MRI - FLAIR
MRI - SWI (Susceptibility)
MRI – T2
MRI – T1 Sagital
Labs
INF DIS/ANTIGEN/MOLECULAR
Adenovirus PCR Quant Plasma
Not Dete
Adenovirus PCR Quant CSF
Not Dete
West Nile PCR CSF
Negative
West Nile PCR Blood
Negative
EBV PCR Quant CSF
Not Dete
VZV PCR Quant CSF
Not Dete
SEROLOGY/INF DISEASE
E Equine Enceph IgG CSF
E Equine Enceph IgM CSF
Calif Enceph IgG CSF
Calif Enceph IgM CSF
St. Louis Enceph IgG CSF
St. Louis Enceph IgM CSF
W Equine Enceph IgG CSF
W Equine Enceph IgM CSF
West Nile Virus IgG CSF
West Nile Virus IgM CSF
Bart henselae IgG
Bart henselae IgM
Bart quintana IgG
Bart quintana IgM
Calif (LaCross) IgG
Calif (LaCross) IgM
E Equine Enceph IgG
E Equine Enceph IgM
St Louis Enceph IgG
St Louis Enceph IgM
W Equine Enceph IgG
W Equine Enceph IgM
Mycoplasma Ab IgG
Mycoplasma Ab IgG Interp
Mycoplasma Ab IgM
Mycoplasma Ab IgM Interp
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
Negative
Negative
<1:128
<1:20
<1:128
<1:20
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
<1:10
0.08
Negative
0.12
Negative
MOLECULAR INF DISEASE
CMV PCR Quant
NEG
Enterovirus RT-PCR
NEG
Epstein Barr Virus PCR
NEG
Herpes Simplex Virus PCR
NEG
Respiratory Viral Panel PCR
Influenza A (subtypes H1, 2009 H1, H3)
Influenza B
Respiratory Syncytial Virus (RSV)
Adenovirus
POS
Human Metapneumovirus
Parainfluenza 1,2,3,4
Rhinovirus/Enterovirus
Bordetella pertussis
Chlamydophila pneumonia
Mycoplasma pneumonia
Coronavirus (HKU1, NL63, OC43 and 229E)
BIOCHEMICAL GENETICS
Phosphoserine
7
Taurine
78
Phosphoethanolamine
0
Aspartic Acid
21
Hydroxy Proline
0
Threonine
304 H
Serine
132
Asparagine
73
Glutamic Acid
58
Glutamine
609
Sarcosine
0
Proline
153
Glycine
339
Alanine
464
Citrulline
9
Alpha Amino Butyric Acid 23
Valine
245
Cystine
57 H
Methionine
42
Cystathionine
0
Isoleucine
70
Leucine
152
Tyrosine
68
Phenylalanine
87
B-Alanine
0
Homocystine
0
Ornithine
80
Lysine
277
Histidine
74
Arginine
146 H
Acute Necrotizing Encephalopathy
(ANE)
What do we know?
Background
Establishment as a new disease in 1995
Higher incidence in East Asian countries
Handful of cases in Caucasians
M=F
Peak at 6-18 months old, but can occur in up to 11yo
< 5yo 81.8%.
Mortality rate 31.8
Neurological sequelae (27.7%)
coagulopathy, hepatic dysfunction, and computed tomographic abnormalities
had a poor prognosis.
Viruses Implicated
Acute Presentation
Convulsions are 1st sign of brain dysfunction
0.5-3 days after onset of antecedent infections
Histology-> encephaloPATHY
Necrosis (due to severe edema) in the thalami,
tegmentum, and dentate nuclei
Florid petechial hemorrhage around small parenchymal
vessels
Patchy cerebral white matter lesions of ANE are not
hemorrhagic
Absence of inflammatory cells in brain parenchyma is
characteristic,
Differentiates ANE from acute disseminated encephalomyelitis &
acute hemorrhagic encephalitis
Pathogenesis
1- Viral invasion of central nervous system
Controversial- via peripheral nerves?
positive viral RNA in CSF and brain but lack of inflammation in brain tissue of fatal cases
Not dependent on infectious agents.
Vascular endothelial cells, astrocytes and neurons -> apoptosis
Viral invasion likely a result not a cause of disease
2- Predisposition
Mutations in the gene Ran-binding 2 (RANBP2) associated with familial or recurrent viral ANE.
Autosomal-dominant ANE due to missense mutations in RANBP2
Hepatic and/or renal dysfunction
3- cytokine storm
proinflammatory cytokines
interleukin (IL)-6, IL-1b, tumor necrosis factor (TNF)-a, soluble TNF receptor
IL-6 level was correlated with worse prognosis
IL-6 and TNF-a -> apoptosis & injury of vascular endothelium, glial cells, and neurons,
-> vascular lesions and breakdown of the blood–brain barrier (BBB)
-> induce brain edema and damage, CNS disorders, and/or systemic symptoms
Other cytokines/chemokines
CXCL8/IL-8, CCL2/MCP-1, and CXCL10/IP-10
Pathogenesis
Investigations - Bloods
Various abnormal findings
Elevation of serum aminotransferases and lactic
dehydrogenase indicates liver dysfunction
Elevation of creatine kinase, urea nitrogen and
amylase indicates concomitant involvement of the
muscles, kidneys and pancreas respectively.
Labs & Diagnostics
Investigations - Imaging
Bilaterally symmetric lesions of the thalami
± lateral putamin & external capsule, tegmentum, cerebellar
nuclei
The lesions are often necrotic and hemorrhagic.
Diffusion-weighted imaging (DWI) -> cytotoxic
edema.
Axial T2-weighted image showing bilaterally symmetric
hyperintensity in the thalami. Note the target
appearance of the lesions.
Axial T2-weighted image showing bilaterally symmetric
hyperintensity in the dorsal pons.
Coronal FLAIR images showing bilaterally symmetric
hyperintensity in the thalami and dorsal columns.
6 Day after 1st MRI
6 Day after 1st MRI
Diagnostic Criteria for ANE
Treatment
Early intervention
improves outcome!
Prognosis
Often grave
Treatment
BACK TO OUR PATIENT
Methylprednisolone 30mg q24hrs.
Mannitol at a 0.5g/kg q6 hour
Serum osm goal ~ 320.
3% hypertonic saline
Na goal high 140 and low 150 range.
NO HYPOTHERMIA PROTOCOL
Patient died on Hospital day 8 (diffuse cerebral edema)
Autopsy: Diffuse brain edema, simplified broad gyri and friable brain
parenchyma consistent with multifocal bilateral hemorrhagic and
ischemic strokes (pending examination after fixation).
Hospital Day 6
Hospital Day 1
Nuclear Medicine Scan
References
1: Neilson DE. The interplay of infection and genetics in acute necrotizing encephalopathy. Curr
Opin Pediatr. 2010 Dec;22(6):751-7. Review. PubMed PMID:
21610332.
2: Wang GF, Li W, Li K. Acute encephalopathy and encephalitis caused by influenza virus infection.
Curr Opin Neurol. 2010 Jun;23(3):305-11. Review. PubMed PMID: 20455276.
3: Mizuguchi M, Yamanouchi H, Ichiyama T, Shiomi M. Acute encephalopathy associated with
influenza and other viral infections. Acta Neurol Scand Suppl. 2007;186:45-56. Review. PubMed
PMID: 17784537.
4: Mastroyianni SD, Gionnis D, Voudris K, Skardoutsou A, Mizuguchi M. Acute necrotizing
encephalopathy of childhood in non-Asian patients: report of three cases and literature review. J
Child Neurol. 2006 Oct;21(10):872-9. Review.
PubMed PMID: 17005104.
5: Mizuguchi M. Acute necrotizing encephalopathy of childhood: a novel form of acute
encephalopathy prevalent in Japan and Taiwan. Brain Dev. 1997 Mar;19(2):81-92. Review.
PubMed PMID: 9105653.