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77.4 Pathology
There is an abscess extending throughout the whole spinal canal from the cervical
to lumbar region. There is compression of the spinal cord. There are marked neurological defi cits. There may be osteomyelitis of the spine.
77.5 Clinical Picture
Fever, pain in the back, neck or all spine. Neurological defi cits from paraparesis to
tetraparesis with sphincter dysfunction. Here the fi rst case reported is abstracted:
A 50-year-old diabetic man presented with fever, leukocytosis, and upper extremity weakness.
77.6 Diagnosis
Physical exam showed weakness of both deltoid muscles 1 out of 5; biceps and triceps 2 out of 5 and weak handgrips bilaterally. MRI holospinal abscess from the
base of the skull to T5 ventrally and dorsally from T1 to L5. Spinal cord severe
compression at C2-C3 and L3-L4 discitis and osteomyelitis.
77.7 Management
• IV antibiotics
• Emergency surgical decompression performed through transoral odontoidecot-
omy, removal of anterior arch of the atlas and partial corpectomy of C2 due to
C2-C3 osteomyelitis.
• Six days later the patient underwent C3-C6 laminectomy and C1-C2 posterior
fusion.
Follow-up showed slow recovery of motion.
The second case was a 45-year-old man with a holocord spinal epidural abscess
involving the whole spinal canal.
Drainage was performed through L2-L3 laminectomy
Two days later the patient developed upper limb defi cits for which C3-C6 and
T6-T9 laminectomies for decompression. A rubber catheter was introduced to irrigate the area between the surgical sites. Slow neurological recovery occurred; the
patient could not be followed-up.
Reference
Lau D, Maa J, Mummaneni PV, Chou D. Holospinal epidural abscess. J Clin Neurosci.
2014;21(3):517–20.
77 Holospinal Epidural Abscess
255
© Springer International Publishing Switzerland 2016
I.M. Eltorai, Rare Diseases and Syndromes of the Spinal Cord,
DOI 10.1007/978-3-319-45147-3_78
7 8
Imported Vaccine-Associated Paralytic Poliomyelitis
78.1 Definition
This is a condition of paralytic poliomyelitis in an unvaccinated individual who
traveled abroad and was in contact with an infant recently vaccinated with oral polio
vaccine (OPV).
78.2 Incidence
This is the fi rst case in the United States since 1999. According to Kelly form
Australia OPV can cause vaccine-associated paralytic polio (VAPP) with a frequency of approximately on case per 2.5 million doses of OPV distributed.
According to the CDC to the mid-1990s approximately 8–10 VAPP occurred annually (Alexander et al.).
78.3 Etiology
Most VAPP occurred in OPV recipients rather than amongst contacts. In the 1990s,
cases of contact VAPP occurred in 1 case per 13 million doses of OPV distributed.
Vaccination was changed to inactivated polio vaccine (IPV) in 2000; thus eliminated VAPP in the United States. From Australia a child received OPV at the age of
4 months and developed immunity to serotypes 1 and 2 but not type 3 and developed
acute transverse myelitis. Poliovirus type 3 was isolated from the stool and the
throat specimens after the onset of myelitis. This was confi rmed by ELISA and
nucleic acid probe hybridization. It is accepted that poliovirus can cause acute
Abstracted from CDC’s publication MMWR ( 2006 )
256
transverse myelitis in 1:125 to 1:800 polio case or 1:300,000,000 to 1:2,000,000,000
doses distributed. Laboratory studies provide plausible evidence of a link between
OPV and VAPP or acute transverse myelitis.
78.4 Pathology
Poliovirus is an RNA virus that is transmitted through the oral-fecal route or by
ingestion of contaminated water. The virus multiplies in the nasopharynx and gastrointestinal tract, from which it spreads to the lymphoid tissue and the blood (viremia). The virus is neurotrophic and settles in the anterior horns and bulbar cells,
whose destruction leads to fl accid paralysis. The incubation period is 5–35 days.
78.5 Clinical Picture
The case reported in the United States:
A 22-year-old woman had not been vaccinated against polio for religious reasons. She went to a study trip to Central America and she was healthy. She was in
contact with the grandchild of her hosts. He was an infant 2 months old; who
received his fi rst dose of OPV 4 days after the woman came to live with the grandparents. After about 40 days she developed fever and general malaise. After 24 h she
got neck and back pains 4 days later she got acute leg weakness and was hospitalized locally and 3 days later was air lifted back to the United States where both legs
were paralyzed and arefl exic and had respiratory failure needing intubation. The
CSF showed high lymphocytic count, high protein, and normal glucose. It was
thought to be Guillian-Barré syndrome and received steroids and plasmapheresis.
Electrophysiological studies showed anterior horn cells or motor axons damage.
MRI showed anterior horn involvement in the cervical and thoracic cord. Serological
studies were positive for Sabin poliovirus type 2 and 3, with no other viruses. She
recovered her respiratory function, but had weakness of both legs.
In the Australian case:
A 6 month old boy developed acute transverse myelitis 7 days after the receipt of
OPV. He was seen age 9 years and was found to have developed immunity to poliovirus type 1 and 2 but not to type 3, which was isolated from his stool and throat
specimens. He later developed immunity to type 3 later.
78.6 Management
Essentially the treatment is supportive:
• care of respiration
• swallowing
• bowel and bladder
78 Imported Vaccine-Associated Paralytic Poliomyelitis
257
Bulbar paralysis need a mechanical management, and anxiety medication to
calm patient may indicate cerebral anoxia, which may precede respiratory problems. Physiotherapy starts in the convalescent stage; orthotics and surgeries in the
chronic stage.
78.7 Prophylaxis
For individuals traveling to an endemic country or where it epidemic they should be
vaccinated. If they were not vaccinated or if it s unknown they should receive two
doses of IPV with and interval of 4–8 weeks and a third dose after 6–12 months.
Individuals who were vaccinated should take a dose of IPV before departure as a
single dose for lifetime. For more details consult the CDC
Reference
CDC. Imported vaccine-associated paralytic poliomyelitis – United States, 2005. MMWR.
2006;55(04):97–9.
Reference
259
© Springer International Publishing Switzerland 2016
I.M. Eltorai, Rare Diseases and Syndromes of the Spinal Cord,
DOI 10.1007/978-3-319-45147-3_79
7 9
Lemierre’s Syndrome
79.1 Definition
Lemierre’s syndrome is caused by an acute oropharyngeal infection with secondary
septic thrombophlebitis of the internal jugular vein and frequent metastatic
infections.
79.2 Incidence
The infection subsided after its description in 1936 when the antibiotics came later;
however, there seems to be recent resurgence. Metastatic infection causing epidural
abscess and spinal cord compression was recently described by Park et al., a manifestation that has not previously been reported.
79.3 Etiology and Pathology
The infection starts in the oropharynx as an acute oropharyngitis caused by the
anaerobic Fusobacterium necrophorum . It leads to postanginal septicemia and sep-
tic thrombophlebitis of the internal jugular vein, from which septic emboli immigrate into the body causing sepsis. The only case described by Park et al., was
complicated by epidural abscess and cord compression.
Abstracted from Park et al. ( 2006 )
260
79.4 Clinical Picture
The only case described by Park et al., was a female subject 43 years old, who had
a sore throat with acute cervical lymphadenitis and fever, for which she received
antibiotic therapy. She developed confusion and weakness of lower extremities 3
weeks later ending in paraparesis. The presentation was fever, tachycardia, agitation
and confusion. She had fl accid paresis of the lower extremities.
79.5 Diagnosis
Diagnosis is based on
1. History oropharyngeal and cervical infections, although many clinicians remain
unaware of this syndrome.
2. Neurological assessment: fl accid paraparesis
3. Lab tests: neutrophilia and raised C-reactive protein (CRP)
4. MRI of spinal cord: showed a collection anterior to the spinal cord from C1-T5
and post-epidural collection from T3-T5 and another collection form T11-L3.
5. CT scan: showed bilateral pulmonary basal consolidation and pleural effusion
also bilateral psoas abscess (all are pyemic infections)
79.6 Management
Laminectomy and anterior C3 foraminotomy, drained the abscess, and antibiotic
fl uconazole, metronidazole, and meropenem therapy continued. Rehabilitation program helped the patient recover movement in 1 year with residual one foot drop.
Reference
Park D, Rezajooi K, Sabin I. Lemierre’s syndrome: an unusual manifestation of spinal infection.
J Bone Joint Surg Br. 2006;88(2):261–2.
79 Lemierre’s Syndrome
261
© Springer International Publishing Switzerland 2016
I.M. Eltorai, Rare Diseases and Syndromes of the Spinal Cord,
DOI 10.1007/978-3-319-45147-3_80
8 0
Listeriosis
80.1 Definition
Listeriosis myelitis is a type of neurolisteriosis caused by listeria monocytogenes,
which is a gram-positive rod bacterium that has a predilection to the central nervous
system (CNS).
80.2 Incidence
Neurolisteriosis affects mostly the immunocompromised subjects. Listerial myelitis
is very rare. A case by Nguyen-Huu et al., and by Josephson et al., were all that were
found in an extensive search of the medical literature, in English and other
languages.
80.3 Etiology
Neurolisteriosis affects mostly immunocompromised subjects, but it also involves
the healthy persons. In the cases of listeriosis infection of the nervous system is
involved. There are 13 serovars of Listeria monocytogenes, the most virulent of
which are serovar 4b, 1/2b, and 1/2a, and cause human and animal disease.
Transmission occurs through the following reservoirs of infection which are soil
and intestinal tract of asymptomatic animals, wild or feral, mammals (cows or
sheep), birds, fi sh, milk, crustacean, urine, and uterine discharge of infected animals. Transmission is through food that is contaminated, whether animal or plant,
venereal transmission, inhalation, transplacental or through the genital tract in labor.
Abstracted from Josephson et al. ( 2006 )
262
Food transmission can be as follows, uncooked vegetables, cheeses, cold cuts, or ice
cream. Listeria is resistant to freezing, heating (1–45°C) and can multiply in refrigerators; spoiled silage favors the bacterial growth.
80.4 Pathology
Neurolisteriosis is more common in patients with compromised immune systems,
but less commonly it affects immunocompetent individuals. The elderly and pregnant women are susceptible and may abort their fetuses. Involvement of the brain is
usually meningitis, meningocephalitis, brain abscess, or hydrocephalus. Spinal cord
involvement is by listerial myelitis, which is rare.
80.5 Clinical Picture
Listeriosis has two different pictures:
• In immunocompromised individuals in debilitated elderly patients and in preg-
nant women. It has a serious picture: fever, septicemia, meningocephalitis, which
may end by brain abscess, or hydrocephalus.
• In healthy individuals, the symptoms are mild. It starts as a fl u like with skin
rash, symptoms of gastroenteritis and may be self-limited as seen in veterinaries.
Myelitis may develop in immunocompetent individuals (Josephson et al.,
Nguyen-Huu et al.). According to the last authors in immunocompromised
patients one in four develops CNS listeriosis. In the cases descried there is
listeriosis myelitis in the cervical region. A case described by Dhiwakar et al.,
had meningocephalitis ending by hydrocephalus and syringomyelia. The clinical
picture is that of pain in the neck and shoulder and upper arm, followed by pare-
sis of the arm and lower extremities, sensory loss was minimal. In one case
hydrocephalus developed due to infl ammatory material in the third ventricle and
was drained.
80.6 Diagnosis
Diagnosis is based on:
1. History: contact with animals or birds or taking contaminated food material
2. Neurological exam
3. Laboratory tests: cerebral spinal fl uid (CSF) will show elevated proteins and
pleocytosis, of the neutrophils, PCR, immunochromatography, immunofl uores-
cence, ELISA, immunomagnetic separation. According to the CDC serology is
unreliable
4. Culture: CSF, cord, or brain biopsies will show the bacteria
80 Listeriosis
263
5. MRI of spinal cord: shows enhancing area in the cord with reenhancing after
gadolinium.
6. rDNA: according to Nguyen-Huu et al., universal Polymerase change reaction
from the biopsy, followed by sequencing of the amplifi ed rDNA gene is the con-
fi rmed diagnosis
80.7 Management
• Steroid therapy for the tissue edema
• Antibiotic therapy:
• 4 weeks of IV ampicillin and IV trimethoprim/sulfamethoxazole (TMP-SMX)
• 8 days of concomitant intrathecal gentamycin
• Surgery:
• Laminectomy and durotomy for decompression and possibly cord biopsy
• Hydrocephalus or syrinx: shunting
References
Josephson SA, Pillai DR, Phillips JJ, Chou D. Neurolisteriosis presenting as cervical myelitis in an
immunocompetent patient. Neurology. 2006;66(7):1122–3.
Nguyen-huu BK, Thümler A, Weisner B, et al. Neurolisteriosis with acute myelitis. Nervenarzt.
2005;76(10):1255–8.
References
265
© Springer International Publishing Switzerland 2016
I.M. Eltorai, Rare Diseases and Syndromes of the Spinal Cord,
DOI 10.1007/978-3-319-45147-3_81
8 1
Melioidosis
81.1 Definition
This is an endemic disease in Sri Lanka causing transverse myelitis due to
Burkholderia pseudomallei.
81.2 Incidence
Although the infection is endemic in Sri Lanka, this is the fi rst case of transverse
myelitis causing paraplegia.
81.3 Etiology
Melioidosis is an infection caused by Burkholderia pseudomallei ; a gram negative
saprophyte in the soil and fi sh water. It is endemic in tropical and subtropical zone
and southeast Asia and northeast Australia. It is underdiagnosed in Sri Lanka, due
to lack of knowledge and awareness of the disease. Also there is lack of diagnostic
tools to confi rm the disease. It has low prevalence. The disease is more common in
males aged 40–60, rare in children and is more common in rural areas (farmers).
The infection is transmitted through the skin inhalation, and ingestion. Predisposing
factors are alcoholism, diabetes, and chronic disease.
Abstracted from Nandasiri et al. ( 2012 )