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Cerebrospinal fluid dynamics in the tardive cauda equina syndrome of ankylosing spondylitis.

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Cerebrospinal Fluid
Dynamics in the Tardive
Cauda Equina Syndrome of
Ankylosing Spondylitis
Christian Confavreux, MD,” Jean-Paul Iarbre, MD, 1
Edouard Lejeune, MD,t Marc Sindou, MD,$
and Gilbert Aimard*
Typical cauda equina syndrome secondary to longstanding ankylosing spondylitis is reported in a 63-yearold man. Radionuclide cisternography demonstrated a
resorption defect of cerebrospinal fluid in the enlarged
lumbosacral dural sac. After transient symptomatic improvement with acetazolamide, a lumboperitoneal shunt
was placed. T h e rate of cerebrospinal fluid, isotope resorption became normal. In the 5 years of follow-up,
partial remission has been observed.
Confavreux C, Larbre J-P, Lejeune E, Sindou M,
Aimard G. Cerebrospinal fluid dynamics
in the tardive cauda equina syndrome
of ankylosing spondylitis.
Ann Neurol 1991;29:221-223
Tardive cauda equina syndrome in ankylosing spondylitis (CES-AS) is a rare but well-known entity since
From the Tlinique de Neurologie, HBpital Neurologique, and $Service de Neurochirurgie, HBpital Neurologique, Lyon, and +Service
de Rhumatologie, HBpitd des Charpennes, Villeurbanne, France.
Received Apr 10, 1990, and in revised form Jun 20 and Aug 8,
1990. Accepted for publication Aug 10, 1990.
Address correspondence to Dr Confavreux, Clinique de Neurologie,
HBpital Neurologique, 59 Boulevard Pinel, 69003 Lyon, France.
Babinski’s pioneer work [l}. To date, 69 cases have
been reported 12-7}. Inflammation and fibrosis predominating o n dura matter and epidural space associated
with enlargement of the caudal sac, narrowing of epidural space, adherence of dura matter to adjacent bony
and ligamentous structures, and bony erosions of laminae and spinous processes are the basic radiological [Z,
81, surgical 13-5, 7, 9-11], and pathological C6, 121
features. The long interval separating the onset of ankylosing spondylitis (AS) and cauda equina syndrome
(CES) and the common finding that AS has been
burned out for several years when CES begins suggests
that active inflammation is not directly involved in its
pathogenesis. Alternatively, retractile fibrosis in the
epidural space could lead t o a “between-trunk-andbark” variety of entrapment syndrome.
We report a patient, with study of the cerebrospinal
fluid (CSF) dynamics by isotopic techniques. A resorption defect of the isotope from the caudal sac was observed. Correction by lumboperitoneal shunting resulted in partial improvement of radicular pain.
Patient Report
A 63-year-old, HLA-B27 positive, graduate engineer suffered from a relapsing form of AS since the age of 23 years.
Sacroiliac joints and spine became fused with pronounced
kyphosis. In 1956, vertebral osteotomy with posterior osseous graft was performed at the T12-L1 level for correction
of the spinal deformation. During the subsequent years, inflammationand pain in the joints subsided and analgesics and
anti-inflammatory drugs could be stopped. The patient never
received radiation therapy to the spinal column for AS.
A new pain in the L-5, S-1, and S-2 distributions began in
1968, at age 51 years, predominantly on the left side, with
severe exacerbations due to mechanical factors. In 1974,
water-soluble contrast medium myelography showed an enlarged and lobulated lumbosacral dural sac with no evident
radicular compression (Fig 1A). Pain worsened progressively
and became chronic and severe. At admission in May 1980,
loss of cutaneous sensation on left perianal region, heel, and
sole, slight weakness on the left in L-5 innervated muscles,
and bilateral ankle areflexia were present. Erythrocyte sedimentation rate was normal. Total protein level was 280 mg/
L, and white cell count was 2 per cubic millimeter in CSF.
Lumbosacral mega cul-de-sac was more apparent on metrizamide myelography as well as erosion of posterior walls of
the vertebral bodies (scalloping) and a posterior lobulated
meningocele predominantly at the L-4 level (Fig 1B). Computed tomographic (CT) scan confirmed erosion of the laminae and posterior spinous processes on their axial side in the
lumbosacral region (Fig 2). Electromyography with needle
electrode examination showed fibrillations and decreased
numbers of motor unit potentials bilaterally in the L-4, L-5,
and S-1 root distribution, consistent with neurogenic abnormalities. F waves were delayed or abolished. Nerve motor
and sensory conductions were slowed in the lower limbs
(30-36 misec).
Noramidopyrine had a partial analgesic effect, but transcutaneous electrical stimulation, carbamazepine, steroids, isaxo-
Copyright 0 1991 by the American Neurological Association
Fig 2 Lumbar computed tomographic scan at the L-4 level. Enlargement of the vertebral canal and defects in the laminae.
Fig 1. Water-soluble contrast medium myelography. (A) 1974.
Enlarged and lobulated caudal sac. (B) 1980. The caudal sac
seems larger with posterior meningocele and vertebra/ scalloping.
nine, and B vitamins were without benefit. In 1983, lightning
pains and chronic crushing pain predominantly in the left
perianal region, heel, and sole became intense. They were
worsened by ambulation, which was restricted to 500 m with
a cane. Clinical examination was unchanged by comparison
with 1780. Radionuclide cisternography was performed with
2 mCi Indium 111 injected at the C-2 level. The cerebral
cisternae and lumbosacral dural sac were visualized within
normal delays (Fig 3A), but isotope was still detectable in the
dural sac at 24 hours. Half-life elimination time of the isotope
at this level was markedly increased (1 1 hours; normal range,
1-4 hours).
Acetazolamide, up to 500 mg per day, reduced pain intensity for only a 6-month period. A lumboperitoneal shunt was
placed in April 1984. Subsequent improvement was pronounced with disappearance of lightning pains, reduced intensity of the chronic pain, and ability to walk up to 2 km.
Neurological examination and dural mega cul-de-sac on C T
scan, however, were unchanged. Radionuclide cisternography was repeated in Qctober 1984. Isotope rapidly diffused
toward lumbosacral dural sac, peritoneal cavity, and bladder
(Fig 3B). No intrathecal isotope was detectable at 24 hours
after injection. Isotope half-time elimination was 1 hour.
During the following years, symptoms fluctuated with periods of intense pain in the legs and inability to walk without
external assistance alternating with periods of complete
symptomatic remission. Radionuclide cisternography was
checked in May 1986 and confirmed that the lumboperito-
222 Annals of Neurology
Vol 27 No 2 February 1771
Fig 3. Indium 111 radionuclide cistwnography. Isotope injection
at the C-2 level. (A) 1983. Prolonged stagnation of the isotope in
the caudal sac at 1 I and 24 hours after isotope injection. (B)
October 1984, 6 months after lumboperitoneal shunt insertion;
rapid dz$fusion of the isotope from the caudal sac to the bladder
through the bmboperitoneal shunt (L.P.D.).
neal shunt was working properly. Since January 1788, pain
has been moderate and ambulation full.
We propose that a hydrodynamic component may be
considered in CES-AS syndrome. Based on the two
myelographies performed at a 6-year interval, the dural
sac increased in size. Acetazolamide benefited the neurological symptoms although only temporarily. Cisternography demonstrated stagnation of isotope inside
the d u a l sac. Lumboperitoneal shunt was able to correct cisternographic abnormalities and, to a lesser
extent, neurological symptoms. CES signs were unmodified, but they had been long-standing and were
probably irreversible.
Defective resorption of lumbar CSF, which physiologically takes place through arachnoid villi in epidural
veins 1131, could contribute to dural sac enlargement.
Furthermore, rapid CSF pressure changes that were
not efficiently damped [14] insofar as the epidural
space was atrophic and the epidural veins were compressed could have contributed to nerve root injury.
According to these data, CES-AS is in some respects
a normal pressure-enlarged dural sac syndrome, especially as opening lumbar CSF pressure has always been
found normal [I, 4 ) in this clinical setting.
CES-AS bony erosion concerns only the inner surface and preserves the outer surface of the vertebral
canal. This feature could be relevant to the hydrodynamic theory, and “erosion” may not be the appropriate term. Owing to the enlargement of the dural sac
and the relative CSF hypertension, a slightly centrifugal
displacement of bone formation during continuous
bone turnover could be postulated.
This dynamic theory needs confirmation. Continuous recording of lumbar CSF pressure in patients with
CES-AS and cisternographic studies performed at the
inflammatory stage of AS before CES-AS onset would
be relevant. In any event, this theory provides a new
approach to CES-AS therapy; early CSF shunting may
be considered for CES-AS prevention.
We thank Dr G. Veillas, MD, Centre de Midecine Nucleaire, Hbpital Neurologique de Lyon, for technical collaboration.
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Brief Communication: Confavreux et al: CSF Hydrodynamics in CES
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syndrome, equine, ankylosis, dynamics, tardive, caudal, spondylitis, fluid, cerebrospinal
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