RECURRENT SPINAL ADHESIVE ARACHNOIDITIS
A C A S E R E P O R T
JAMES PITÁGORAS DE MATTOS * — CHARLES ANDRÉ * BERNARDO ANTONIO MURTINHO COUTO *
S U M M A R Y — S p i n a l a d h e s i v e a r a c h n o i d i t i s i s n o t a n u n c o m m o n d i s e a s e , u s u a l l y h a v i n g a m o n o p h a s i c c o u r s e . W e s t u d i e d a n a t y p i c a l p a t i e n t w i t h r e c u r r e n t s p i n a l a d h e s i v e a r a c h n o i d i t i s n i n e y e a r s a f t e r i n t r a t h e c a l a n e s t h e s i a a n d t h e f i r s t a t t a c k o f t h e d i s e a s e . A l s o n o t e w o r t h y w a s t h e f a v o r a b l e e v o l u t i o n a f t e r s u r g e r y .
Aracnoidite adesiva medular recidivante: registro de caso.
R E S U M O — A r a c n o i d i t e a d e s i v a m e d u l a r n ã o é d o e n ç a i n c o m u m e h a b i t u a l m e n t e a p r e s e n t a - s e c o m c u r s o m o n o f á s i c o . E s t u d a m o s o c a s o a t í p i c o d e p a c i e n t e c o m a r a c n o i d i t e a d e s i v a m e d u l a r r e c i d i v a n t e n o v e a n o s a p ó s r a q u i - a n e s t e s i a e d o p r i m e i r o e p i s ó d i o d a d o e n ç a . T a m b é m é d i g n o d e n o t a a e v o l u ç ã o f a v o r á v e l q u e s e s e g u i u a p ó s c o n d u t a n e u r o c i r ú r g i c a .
Spinal adhesive arachnoditis ( A S A ) affects preferentially adults of both sexes, most often following intraspinal use of various agents, being thus an important medical social problem. Its course is usually monophasic either progressing unrelentlessly or stabilizing, and even recovering to a variable degree. W e report a case with atypical evolution, consisting of prolonged remission followed by a second subacute attack nine years later.
C A S E R E P O R T
A Caucasian man 33 years old had an unremarkable clinical history until a g e 24. One day after being submitted to intrathecal anesthesia f o r a postectomy, h e developed progressive paraparesis, fecal and urinary incontinence and sexual impotence. F o u r months later he was paraplegic. A myelographic diagnosis of A S A was made, and t h e patient w a s treated with 21 cisternal injections of Depo-Medrol ( R . ) followed b y prednisone in low oral doses, without benefit. The course remained stable f o r s i x months, followed b y slight bilateral arm weakness. T h e patient then started an intensive physiotherapy program. D u r i n g the following year he recovered almost to normal, regaining muscular power and sphincteric continence. T h e patient g o t married and had a child, n o w 19 months old. H e w a s doing quite well until one month before admission, when sphincteric incontinence, sexual impotence and tetraparesis of rapid evolution all recurred, along with paresthesias around the cervical column. Several lumbar punctures w e r e tried unsuccessfully, and the patient w a s treated with parenteral dexamethasone, without benefit. Past medical history disclosed hilar tuberculosis during adrenocorticosteroid therapy, which w a s discontinued, four years ago. T h e patient was treated successfully with para-aminosalicylic acid, streptomycin and isoniazid for one year. Three years ago, he had acute viral hepatitis, with total resolution and
* I n s t i t u t o d e N e u r o l o g i a D e o l i n d o C o u t o , F e d e r a l U n i v e r s i t y o f R i o d e J a n e i r o .
without sequellae. General physical examination was unremarkable. Neurological examination disclosed, at inspection, shoulder-girdle fasciculations and amyotrophy, specially of the left limbs. There were also bilateral foot drop and left side hypothermia. Upright posture was possible only with aid, and supported gait was pareticataxic. There w a s tetraparesis, with muscular power more markedly reduced in distal segments and at the right side. Tonus was increased in both legs and the right arm, and reduced in the left arm. T h e four limbs were ataxic, with obvious deterioration after eye closure. Superficial reflexes were abolished, and Babinski sign was present bilaterally. Muscle stretch reflexes were brisk at the right side and abolished at the left one. Patellar and ankle clonus were readily
elicited at the right side. Cutaneous sensation was altered (Fig. 1 A ) . Deep sensation was bilaterally reduced in the lower limbs. T h e re were fecal and urinary incontinence. Cranial nerves and mental status were normal. Syphilis screening tests were negative as were normal routine studies, except for mild pyuria and hematuria. Cerebrospinal fluid ( C S F ) obtained b y cisternal puncture revealed 1.4 cells and total protein level of 13mg per mm3, with negative V . D . R . L . . and globulin qualitative tests. Radiological examination of the cranium, vertebral column and thorax was normal, except for the presence of minute residual amounts of liposoluble contrast agent in the cervical column and at the base of the skull. Cisternal descending and lumbar ascending myelographies showed total obstruction to contrast flow at the mid-cervical and mid-thoracic levels respectively (Fig. 2 ) . Partial obstruction and typical candle-guttering appearence were also present in a lumbar myelography done nine years before, specially at the thoracic level.
W i t h an assumed diagnosis of A S A , and considering the rapid deterioration and total C S F block, the patient w a s submitted to cervical laminectomy from C3 t o C5 with extensive
C O M M E N T S
The introduction of spinal anesthesia in the first half of this century was soon followed by experimental studies and case reports of its potential neurological complications. Among these, A S A stands out as a potentially preventable disease 9, usually related to either direct toxic effects or late immune reaction to the administered
drugs- Causal agents may be the different anesthetics per s e2
, contaminant residua of detergents used in the sterilizing process of syringes and vials of anesthetic
solutions3
, or a synergistic action of both substances 6.
Surgical management of spinal adhesive arachnoiditis is restricted, as in this case, to disease of rapid evolution, specially in the presence of signs of a progressive
lesion in a limited region with documented spinal block 7
. Systemic adrenocorticosteroids are also, of controversial value, but widely used in cases of recent onset and rapid evolution. Obviously, its intrathecal use following the first attack was ineffective, if not harmful. There is increasing evidence that these drugs may cause potentially
serious meningeal inflammation!.8
. Long-action drugs of this class possess poly-ethylene glycol and long-chain fatty acids, thought to be responsible for this effect 5. The role of physiotherapy in promoting a favorable evolution is difficult to evaluate, although it was an important factor in maintaining the motivation and patient commitment to treatment.
This report of a single case of A S A is justified by its atypical, biphasic course with long remission, which was never found among patients previously admitted to
our institution. The monophasic evolution of the disease is well documented 3
,1 0
. It may either progress steadily or remit with sequella of variable degrees. In a study of the
radiological manifestations of the disease, Lombardi et a l .4
disagree with this general view, specifically quoting many cases of remission, even for long periods. Meanwhile, they do not report these cases and paradoxically mention the serious evolution of their ten nonsurgical patients. Further, they specifically excluded the postanesthetic group of patients from the study. Anyway, this case report should attract attention of neurologists to our poor knowledge of the natural history of remitting A S A . W e probably should be a little more cautious in stating the long-term prognosis of patients who have an initial good outcome, until studies are made which specifically address this matter.
R E F E R E N C E S
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