We present the case of a 22-year-old man who was diagnosed with tonsillitis and treated with antibiotics. Although the symptoms subsided, 1 week later, he presented with weakness in the lower limbs and was hospitalized. The weakness in the lower limbs worsened; he developed difficulty speaking and was transferred to our hospital. Laboratory tests showed a white blood cell count of 10,600/
Epstein-Barr virus (EBV) is a deoxyribonucleic acid (DNA) virus in the herpesvirus genus. This virus may be associated with the onset of infectious mononucleosis and lymphoma [
Acute disseminated encephalomyelitis (ADEM) is an acute demyelinating disease resulting from inflammation and autoimmune targeting of the central nervous system. This disease is more common in children than in adults [
We report herein an adult case of ADEM triggered by EBV infection.
A 22-year-old man with no relevant medical history developed a sore throat and fever. He was diagnosed with tonsillitis and was started on treatment with antibiotics. Thereafter, the symptoms subsided, but he experienced weakness in the lower limbs 1 week later. He, therefore, visited another hospital and was admitted. He was later transferred to our hospital after exacerbation of the weakness in the lower limbs and development of difficulty speaking.
The patient was afebrile, with a blood pressure of 134/76 mmHg and a heart rate of 75 beats/min. Neurological examination revealed difficulty speaking and weakness of the iliopsoas muscle. No involvement of the cranial nerve system was identified, and reflexes remained intact. No ataxia was evident.
Laboratory tests (Table
The patient’s laboratory test results.
Day of admission | 14 days after admission | Reference range | |
---|---|---|---|
White blood cells (/ | 10600 | 10200 | 4000-9000 |
Lymphocytes (/ | 2330 | 1820 | |
Lymphocyte (%) | 22 | 17.8 | 26.6-46.6 |
Atypical lymphocytes (/ | 2540 | 0 | |
Atypical lymphocyte (%) | 24 | 0 | 0-3.0 |
AST (IU/L) | 26 | 24 | 13-33 |
ALT (IU/L) | 41 | 67 | 8.0-42 |
| 29 | 22 | 11-47 |
Total bilirubin (mg/dL) | 0.4 | 0.6 | 0.2-1.2 |
Albumin (g/dL) | 3.8 | 3.8 | 4.0-5.0 |
BUN (mg/dL) | 13.1 | 15.3 | 8.0-22 |
Creatinine (mg/dL) | 0.58 | 0.63 | 0.6-1.1 |
CRP (mg/dL) | 1.39 | 0.02 | 0-0.19 |
Anti-EBV VCA IgG (times) | 160 | 80 | <10 |
Anti-EBV VCA IgM (times) | 20 | 20 | <10 |
EBNA (times) | <10 | <10 | <10 |
Anti-EA-DR IgG (times) | <10 | <10 | <10 |
Abbreviations:
Both IgM and immunoglobulin G (IgG) antibodies against EBV-viral capsid antigen (VCA) showed positive results. Levels of EBV-nuclear antibody (EBNA) were below the level of sensitivity, as were levels of immunoglobulin G for EBV-early antigen-diffuse-type and restricted-type antibodies. No elevation was found by comparing paired sera for IgG antibody against EBV-VCA during the onset and recovery phases. However, there was positivity for IgM antibody against EBV-VCA and EBNA negativity; thus, the diagnosis was EBV infection [
In peripheral blood, EBV was confirmed using real-time polymerase chain reaction (PCR). There were
At the time of admission, cerebrospinal fluid revealed the following: white blood cell count, 46/mm3 (97% monocytes and 3% polymorphic neutrophils); protein, 71 mg/dl; glucose, 70 mg/dl; and negative results for EBV-DNA. Ten days after admission, cerebrospinal fluid analysis revealed the following: white blood cell count, 21/mm3 (100% monocytes); protein, 44 mg/dl; and glucose, 57 mg/dl.
At the time of admission, magnetic resonance imaging (MRI) of the spine revealed a hyperintense lesion at the Th11 level of the lower spine on T2-weighted imaging (T2WI). No contrast effect was seen. MRI of the brain at the time of admission showed hyperintensities on the right cerebral peduncle, bilateral thalami, posterior leg of the left internal capsule, and right corona radiata on T2WI and fluid-attenuated inversion recovery (FLAIR) imaging (Figures
(a) Sagittal MRI T2-weighted imaging (T2WI) of the spine at the time of admission showed a hyperintense lesion in the spinal cord at the Th11 level of the lower spine (arrow). (b) Axial MRI T2-weighted imaging (T2WI) of the Th11 level of the spine at the time of admission showed a hyperintense lesion in the spinal cord (arrow).
(a) T2WI imaging of the brain showed a hyperintense lesion at the right cerebral peduncle (arrow). (b) FLAIR imaging of the brain showed a hyperintense lesion at the bilateral thalami (arrow). (c) FLAIR imaging of the brain showed a hyperintense lesion at the left internal capsule (arrow). (d) FLAIR imaging of the brain showed a hyperintense lesion at the right corona radiata (arrow).
No obvious abnormalities were evident from electroencephalography performed on day 5 of hospitalization.
Hepatosplenomegaly was observed on abdominal ultrasonography, with diameters of 15 cm for the liver and 11 cm for the spleen.
We diagnosed infectious mononucleosis because he had acute tonsillitis, fever, and the presence of atypical lymphocytes. Furthermore, we diagnosed infectious mononucleosis due to EBV primary infection since the results showed positivity for IgM antibody against EBV-VCA and negativity for EBNA, although no elevation was found by comparing paired sera for IgG antibody against EBV-VCA during the onset and recovery phases.
Furthermore, we suspected ADEM-associated EBV based on the course of onset, the appearance of atypical lymphocytes, and results from MRI of the lower spine and brain and initiated steroid pulse therapy (methylprednisolone at 1,000 mg/day) for 5 days.
The patient showed improvements in speech from the day after starting steroid pulse therapy. After steroid pulse therapy had been performed for 3 days, other symptoms showed improvement. Communication with the patient normalized and steroid therapy was gradually reduced.
A follow-up brain MRI 12 days after admission showed the disappearance of the initial lesions (Figure
(a) T2WI imaging of the brain 12 days after admission showing the disappearance of the initial lesion (arrow). (b) FLAIR imaging of the brain 12 days after admission showing the disappearance of the initial lesion (arrow). (c) FLAIR imaging of the brain 12 days after admission showing the disappearance of the initial lesion (arrow). (d) FLAIR imaging of the brain 12 days after admission showing the disappearance of the initial lesion (arrow).
EBV is known to cause neurological complications, including meningitis, encephalitis, cranial nerve palsy, myelitis, peripheral neuropathy, and Guillain-Barré syndrome [
MRI has been reported as a useful diagnostic method. This modality shows multiple, disseminated lesions and white matter lesions [
Various diagnostic criteria for ADEM have been proposed [
Direct infiltration of the virus into nervous tissue [
The treatment of ADEM is empirical, and high-dose corticosteroid (1 g/day) for 3–5 days has been reported to be useful [
In conclusion, we encountered a case of adult-onset ADEM with EBV. This pathology is rare and difficult to diagnose, but careful diagnosis is crucial since appropriate treatment is necessary to improve symptoms,
Epstein-Barr virus
Deoxyribonucleic acid
Acute disseminated encephalomyelitis
Magnetic resonance imaging
T2-weighted imaging
Fluid-attenuated inversion recovery.
Our submission is a case report. So, the datas used to support the findings of this case report are included within the article. No extra data was used to support this study.
The authors state that they have no conflicts of interest.