experience in the surgical management of spontaneous...

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Experience in the surgical management of spontaneous spinal epidural hematoma Depar?ments of Neurosurgery and Diagnostic Radiology, Chang Gung University and Chang Gung Memorial Hospital; and Department ofPhysica1 Medicine and Rehabilitation, Taoyuan General Hospital, Tmyuan, Taiwan Object. Sporrtaneous spinal epidural hematoma (SSEH) is a rare disease entity. Althwgh m y cases have been mpmtd in the litmatwe, controversy persists as to its origin, diagnosis, and timing of tr#rtmeot Tbe authors conduct- ed a study in patients treated in their hospital and report the rwruits. Methods. Clinical data obtainad in 35 patients with SSEH wen rctmpedvely =viewed. Age, sex, hrstory of hyper- tension, and history of anticoagulation therapy were recorded, and &a wue analyzed to clarify thc possible predis- posing factors of SSEH. Neurological outcomes were reappPaiscd wing a staaQardizad gdmg system and correlated with the time interval from initial ictas to surgery, duration of compk aeurologd deficits, tW the rapidity of dete- rioration of jwalysis. Nonpmmetik methods and Spmmm ranlr-curmw toe- wwe wed for staristical analysis. C01u:Iuswns. SutgGipr is a sak and effective promhe b heat SSEH. n# ~~~ mortality rate was 5.796, the surgery-related ampkdon rate was 2.%, and then w- no opcmtha-idatd deaths. Ndogicd outcrmnc after surgery is positively obfialrttd with W v e wudugid ddkita (88.9% mriplco~ nxmrey in patients with - ddicits compand with 37.5% in ttiose with coqiete Mcita Ep < 0.0011). Zn p&ents in whom tbtrimsiow~initiai~ww~(<48~>~io~Qdmrtiosofc~~sy toms was also briefer (< 12 haw), thm is a positive cornlation with better nemhgid and functiortal -very (p < 0.05). S PONTANE~ spinal epidural bentataara is an uncom- mon but disabling disease entity. Holtas, ct d.;LO estimated the incidence of SSQ? to be 13 hlmdividu- in a population of 1.49 million, or 0.1 patients per fW),000 individuals. Although appmxkmtcfy 400 cases have been mported in the litua~e,'~-~4~~* operative seTits with long-term clinical follow-up data r& few.I- The classic symptoms of SSEH are usually ini- tiated with acute back or neck pain mud the involved vertebrae witb pain radiating to the corresponding der- matomes. ?he sip and symptoms of rapidly evolving neural element corqmsshn develops minudes to days If left untreated, f k p m d y complete and permanent al deficits caa axxu d death has been occa- y nporrteda6 Although cases of successful nonoper- atiw merit have been rqmtd, wid diagnosis and ploajpt csrSirpation of the spinal epkitml clot remain the standard ~ e m e n b ~ Previous sugkd reports, howev- er, usually lack standard documtati011 of loag-term neu- Abbreviations used in this paper: ASIA = Amxican Spinal Injury Association; CI confi&nce interval; CI' = computerized tumopphy; MR = magnetic mmarm; mRS = modifkd Rankin Scale; SSEH = spontaneous spinal ep&d hematoma rotom or they arc Based on a heterogeneous patieat ptpdath that might bias the statistical analysis.22 In tk present study we nwiewed the m81fqamnt-dated dataobtainedin~~withSSEH~atowinstitu- tion. We analyzed preopedve clinicat presentations with associated predisposing factors as well as tbc nlationships between the FMeOPefafive time intervals and postopera- tive results. Thc pqxm of this study was to provide more sufficient informafin to clanfy this surgically curable disease. The charts and mdbpphs of patients with epidural spinal hematom8 treated in the aeuromgical department of Clung Gung Mef~)ial HoqM dunng a rtcent 17- year Mod were retmpctively reviewed. T3e definition of nmttaumatic spinal epidural hk-m proposed by Lonjon, et al.,% was used to sckct and homogenize the patient population. Cases involving spinal hematomas developing affcr verttbraI trauma or after iatrogenic pro- cedures were not included in the study. Tlmty-five pa- tients (26 males and nine females) met all criteria and form the basis of this study. The size of SSEH was con- J. Neurosurg: Spine / Vdurne 100 / January, 2004

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Page 1: Experience in the surgical management of spontaneous ...dlweb01.tzuchi.com.tw/DL/AcdActive/content/research/document...Experience in the surgical management of spontaneous spinal epidural

Experience in the surgical management of spontaneous spinal epidural hematoma

Depar?ments of Neurosurgery and Diagnostic Radiology, Chang Gung University and Chang Gung Memorial Hospital; and Department ofPhysica1 Medicine and Rehabilitation, Taoyuan General Hospital, Tmyuan, Taiwan

Object. Sporrtaneous spinal epidural hematoma (SSEH) is a rare disease entity. Althwgh m y cases have been mpmtd in the litmatwe, controversy persists as to its origin, diagnosis, and timing of tr#rtmeot Tbe authors conduct- ed a study in patients treated in their hospital and report the rwruits.

Methods. Clinical data obtainad in 35 patients with SSEH w e n rctmpedvely =viewed. Age, sex, hrstory of hyper- tension, and history of anticoagulation therapy were recorded, and &a w u e analyzed to clarify thc possible predis- posing factors of SSEH. Neurological outcomes were reappPaiscd wing a staaQardizad g d m g system and correlated with the time interval from initial ictas to surgery, duration of compk aeurologd deficits, tW the rapidity of dete- rioration of jwalysis. Nonpmmetik methods and Spmmm ranlr-curmw toe- wwe wed for staristical analysis.

C01u:Iuswns. SutgGipr is a sak and effective promhe b heat SSEH. n# ~~~ mortality rate was 5.796, the surgery-related ampkdon rate was 2.%, and then w- no opcmtha-idatd deaths. N d o g i c d outcrmnc after surgery is positively obfialrttd with W v e wudugid ddkita (88.9% mriplco~ nxmrey in patients with - ddicits compand with 37.5% in ttiose with coqiete Mcita Ep < 0.0011). Zn p&ents in whom t b t r i m s i o w ~ i n i t i a i ~ w w ~ ( < 4 8 ~ > ~ i o ~ Q d m r t i o s o f c ~ ~ s y m p toms was also briefer (< 12 haw), thm is a positive cornlation with better nemhg id and functiortal -very (p < 0.05).

S P O N T A N E ~ spinal epidural bentataara is an uncom- mon but disabling disease entity. Holtas, ct d.;LO estimated the incidence of SSQ? to be 13 hlmdividu-

in a population of 1.49 million, or 0.1 patients per fW),000 individuals. Although appmxkmtcfy 400 cases have been mported in the l i t u a ~ e , ' ~ - ~ 4 ~ ~ * operative seTits with long-term clinical follow-up data r& few.I- The classic symptoms of SSEH are usually ini- tiated with acute back or neck pain mud the involved vertebrae witb pain radiating to the corresponding der- matomes. ?he s i p and symptoms of rapidly evolving neural element corqmsshn develops minudes to days

If left untreated, f k p m d y complete and permanent al deficits caa axxu d death has been occa-

y nporrteda6 Although cases of successful nonoper- atiw merit have been rqmtd, wid diagnosis and ploajpt csrSirpation of the spinal epkitml clot remain the standard ~ e m e n b ~ Previous s u g k d reports, howev- er, usually lack standard documtati011 of loag-term neu-

Abbreviations used in this paper: ASIA = Amxican Spinal Injury Association; CI confi&nce interval; CI' = computerized tumopphy; MR = magnetic mmarm; mRS = modifkd Rankin Scale; SSEH = spontaneous spinal e p & d hematoma

r o t o m or they arc Based on a heterogeneous patieat ptpdath that might bias the statistical analysis.22 In tk present study we nwiewed the m81fqamnt-dated d a t a o b t a i n e d i n ~ ~ w i t h S S E H ~ a t o w i n s t i t u - tion. We analyzed preopedve clinicat presentations with associated predisposing factors as well as tbc nlationships between the FMeOPefafive time intervals and postopera- tive results. Thc p q x m of this study was to provide more sufficient informafin to clanfy this surgically curable disease.

The charts and mdbpphs of patients with epidural spinal hematom8 treated in the aeuromgical department of Clung Gung M e f ~ ) i a l H o q M dunng a rtcent 17- year Mod were retmpctively reviewed. T3e definition of nmttaumatic spinal epidural hk-m proposed by Lonjon, et al.,% was used to sckct and homogenize the patient population. Cases involving spinal hematomas developing affcr verttbraI trauma or after iatrogenic pro- cedures were not included in the study. Tlmty-five pa- tients (26 males and nine females) met all criteria and form the basis of this study. The size of SSEH was con-

J. Neurosurg: Spine / Vdurne 100 / January, 2004

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Spontaneous spinal epidural hematoma . . --

s i d e d to comprise the number of vertebral segments it spanned. The distribution of SSEHs was divided into cer- vical, cewicothoracic, thoracic, thoracolumbar, and lum- bar areas. Diagnosis of S S m was confirmed by intraop- erative and pathological examinations. To &fine the px&sposing factors of SSEH, attention was paid to any action associated with strainiug during the initial onset of symptoms, including defecation, voiding, vomiting, sneezing, coughmg, Valsalva maneuver, lifting objects, bending the body forward, and turning while in bed. Blood pressure rates were d e d in the emergency &- p w e n t and during the posfopeiative course; however, only in the latter was the measurement considered mean- ingful for diagnosis of hypertension. A chgnosis of hy- pertension was based on the following criteria: systolic/ diastolic blood pressure at least 14U/90 mm Hg or the need for antihypertension

Neurological Recovery

Neurological recovery was reappraised using the five- tiered ASIA gradrng sy~tem.~ Motor staOus was nxoded at the time of surgery and at 1 month* 3 months, 6 moadu, and 1 year postopemtively. For the purpose of statistical analysis, the alphabetical ASIA scorts were assigned nu- merical grades: A, 1; B, 2; and so on. Thc S S S H + W mortality was assigned a grade of 0, and was incorporated

tied ASIA scores fw statistically analysis. The '""r S (Table 1) was used to evaluate the p o s t o ~ v e functional status.* In cases naiviag an mffS score of 1, or 2, the patients were considered to have ma& a func- tional recovery.

Statistical Analysis

Statistical analyses were perf& using a commer- cially available software package. Nonpm- tests were usad to analyze the correlations between the vari- ables of the characteristics of F e n t s or SSEH, preopera- tive time intarVal of sympaonrs, md the s u g e q ~ t e d waxme (ASIA grades) st 1,3,6, and 1 2 n x a h s ~ o p - erativeiy, For all statistical tests, a pmbddity value less than 0.05 ( twWed) was c o n s i ~ signifbmt.

Rt!s& Patient Population a d Pr-ILS oj'SSEII

Thirty-five (26 male and nine female) patients fblf5lled the criteria of SSEH; their ages m g d froI1.17 months to 77 years ( mean age 42.6 2 3.8 years) (Fig. 1). In 12 patients (34.3%) h-im was Qcummted in emergency writ, ~~ 've hypertension was docu- men& in only s e v e n V ( 2 0 % ) . One patient (29%) took warfarin (10 mg/day) to pvent hrmbosis of the artificiat aortic valve and the i n h e r n a h d xxmdbd ratio

time was 1.94 in the emergency depart- EH. Three patients (8.4%) rcceivd tow-dose aspiria for a *mote c&vw- addent igIhmbL Only one patient (2%) sugered

mild t h r o m ~ (102 X lV/dl) in the emergency department, but p@cw count spontaneously recovered to 200 X lP/dl 1 week 'vely. Nineteen patients (54%) experienced subj-g-awciatd events

TABLE 1 Swnmary of the rnRT

Score Dtscription

0 no symptoms 1 no signrfmat dipabitity despite symptom: able to perform all

usuai duties & activities 2 slight disability: unabkto pMfwm all previous activities; able

to care fix own affairs w/o assistance 3 moderate cbbility: quires some help, but able to walk w/o

assis~ance 4 umckmtdy evere disability: unabk to walk w/o assistance or

attgdtobodilynads 5 x v e disability: bedridden, incontinent, & requires constant

nursing care & attention

during the initial symptom attack. --three patients (94%) expMienced sudden+nset back or neck pain includ- ing a 7--4 hbt, who presented with irritable cry- ing, and a 2-year4d ch&i, with tonic po- when turning the heid to the I& other two patients (5.7%) with lumbar SSEN suilkqd sudden f d muscular weakness after chm& lour-beck paie The orientations of the SSEHs idudcd five atrterior (14.396). three lateral (8.6%), nine postemlatad (25,7%), and 18 hematomas posterior to the dural sac (51.4%). Overall the SSEHs extended over one to 10 v.mebd !a@mmts (medirtn four segments [95% CI

) 2). ?gbk 3 provides a sum- areas and associatad clinical

neurulogical m. F i p 2 provides a g q h c rep- ~~ of all b d v e d segments. Reoperatrve deficits were incomplete in 18 patients (5 1.7%) and complete in 1 7 (48.3%). Reopetative ASIA gtacBes and neurological de- ficits m suamarhd in Table 4. In each F e n t with incomplete neurological deficits, the power of portions of key muscles was no greater thgn Grade 2 and the duration of symptoms raqed fkom 10 hours to 1 month.

Radiobgictal Diagnosis

Before 1994, SSEHs were diagnosed in five patients by myelography and in 12 nts by pstmyeIography CT scanning. After 1994, maghag w ~ w c d to establish a diagnosis in 18 patients. In 12 cases myelography dem- onstrated complete block of contrast at the lowest level of

Ast FK;. 1. Bar graph demonstrating distrhtion of age (in years) of

35 patients with SSEH.

J. Neurosurg: Spine / Volume 100 / JunmTry, 2004 39

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C. C. Liao, et al.

TABLE 2 Demographic d m and the location of SSEH in 34 prrtiarrts

LocationdSSE)I - -

VarioMe Catplical CkdWaacic Thoracic 73~~a~dumbar Lumbrrt Total

-

* Vdues an uy#tssed as the mesorr 2 standard deviations.

SSEH, and in five it revealed spbde-shaped epidural fill- ing defects. On p o s t m y e l o ~ y CT scam of the spine, SSEHs wee ~~ as iatmcmd sofc-tissue densi- ties that wae relatively h ' to spinal cord and hypoin- to a MR images, tht SSEH af3pm?&a 0 9pzndle-zi- iBtmcad space- occupying soft-tissue mas. Comprred with rnycbgraphy

tbebARimqbgwasabletodeliaeate the amin==?? full extent o the epidural clot, except in the Zyear-old child in whom the SlFEH extended fbm 42-2 to T-4. Nwrologd r ~ ~ ~ y e r y in patients with MR kna@ag-doc- m t e d dbpxes, however. was lot s i g d W d y differ- ent from that in thost with CT rnyekqpgdxy- ar tnytlog- *y alme-b@ ciiaga- ( h a m - U-test, p 3 0.26). On T,-weighted MR images, the SSW s i g n a h w m hornog~usly isointRnse to the spud wrzi in 15 patha (Fig. 3) and hyperintense in five patients pig. 4). Un T,-weieted images, SSEW signals showed htaeroge- news intensity: 16 pnmanly hyprk@nse, me i andoseh~tcmetothespiaadcord tratbn, ~~ enhancement surro-g the dd was demonsW in four cases, in none of which was en- h-nt homogeneous or intense. On T,-weighted im- ages ebbed in e a t patients, we abserved bypaintense signal in the compressed spinal cord representing intra- medullary edema (Fig. 4). Fwr (50%) of the eight patients with cord edema and o m (10%) of the 10 withait cord &ma did not experience hctional zecovery by the time of the last recoded examination. Although neurolqpcal recovery seemed poorer in the patients in whom there was an intmmedullary hyperbtense signal on T,-wergftted im- ages, the difference. was not sigMfimt to draw a amclu- sion about the mlationship betwm cord edema and goor neur010gical recovery (Fisher exact test, p = C1.12), Four *ts underwent angiqpptry. In one case & m y wtrs perfbmd befm the operation bet- fkquont flue- tuati;ons of complete neurological deficits were followed by rap'd recovery in 3 days, which made physicians sus- pect tk possibility of varnrtar lesion. Thee patients mienvent postoperative mgmgqhy because &cult hemostasis was noted intraopedytly. In all cases, how- ever, the tests proved negative for vascular ancmabs.

Surgical Treatment and Ourcorne

One patient did not undergo the operation. and his data were excluded from the analysis of surgical outcome. He recovered from the 4-hourduration complete spinal cord dysfinction spontaneously, and follow-up MR imaging

performed 5 days after symptom onset revealed that the thoracic SSEH had resolved. In the five cases of anterior- ly loc- SSEHs, one patient miemmt anterior cervical d i s e c ~ y aad evacuation of the short clot at 0 - 6 , but tho IWH&&$ four required bmhectomy because the clots cove&. mvad segmenfs (&u leveb) and were

rior wen a1 epichid clots. follow-up pariod nqed from 3 months to 15 p a s {mean 4.32 i- 0.59 pars). There was neither ~-~ death nor reammt himtoma. A s u r g e r y - W complication of wound disruption oc- cumd in a 2-year-old boy (2.9%). but the lseurological o u ~ w a f ~ 6 m o n t h g ~ ~ o p e r a o i o n ( f r o r n an A S U grade of A ta one of E). Ibe interval m e e n symgtam ewet @aia Mdlor ~~ &kit) to oper- ~ ~ ~ 1 0 h o u r s t o 2 m o n d r s ~ ~ 2 8 h o u r s ~ 9 5 % CI m.3-~n.6 h o ~ ] ) , status de- m i m t t x i m a w y a u c w e r v P m o f t i w : minutest02hoursin l&patients,2t024ho~inninepa- tients, and longer tbaa 24 hours in seven patients. The praoperative duration of maximal d o g i c a l deficits mgcd from 0 bouts (symptom in programion) to great- er than 1 month (madian 12 hours [%% CI 13.7-93.7 housl). In the 28 m g i d y tmattxi patients with incorn- piete mumlogical deficits, status in 14 (77.8%) recovered to an ASIA grade of D within 1 month after operation, in 16 (88.996) to a p d e of E within 1 year, and all pafients by the time of last reamled follow up (mean 4.03 f 3.48 years). In 16 surgicaily treated patients with complete mu- rdogical deficits, status in six patients (373%; five by 1

t

TABLE 3 Tllre Iocaiion of SSEHs and mlotcd p~opemtive

neurologicai dtficits

-Y M* w- Brown- orCsudr

Involved A m atby pkgia SOquard Equhra T d

anicat 4 4 1 9 a.%'vi- 7 1 8 thorock 9. 9 thoracokunbar 6 6 lumbar 1 2 3 dMal 273 4 2 2 35*

*Includhg the patient with spontaneous n c o v a y in hours witbout oper- ative aatmcnt.

J. Neurosurg: Spine / Volume I00 / January, 2004

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Spontaneous spinal epidural hematoma . --

TABLE 4 Preopemtive neumlogical deficits and tfrc nlased ASIA grades

WP Radicu lopathy ASIA Myelop Hemi- Brown- or Grade *Y plegia SCquard CIudP Equina Total

A 17* 17* B 3 3 C 5 3 8 D 2 1 2 2 7

total (5%) 27 (77)* 4 (1 1.4) 2 (5.7) 2 (5.7) 35* V& l m l * Includes the one case in which neurological Mkits mvet.ed sponta-

FIG. 2. Bar graph showing distxibutim of involved segments of neously without operation. SSEH in 35 patients.

year and one by 2 years) recovered to an ASIA grade of E after a mean follow-up period of 4.88 & 3.46 years (Table 5). The follow-up periods of patients with complete and incomplete preoperative neurological deficits w e n not si@caatly different (Mann-Whitney U-test, p = 0.4). Ne~010gid llccovery in patients with inmmpkte preop erative deficits, however, was si@amtly better than that in those with complete deficits (M-Whitney U-test, p < 0.001).

In patients with complete preoperative netmEogrcal def~its, three time factors (interval from initial to operatiun, rate of symptom pmgmsion, and duraticn qf maximal deficits) were analyzed with the final surgery- rtlatad outcums in terms of the ASIA gmhg system. T b e ~ was no correlation between outcome and the rate of symptom progression. The interval betwen initial onset to opmt~on and the duration of maximal deficits were inversely cmiated with the W neurowal recove- ry reflected by ASIA grades (Spearman rank-amehion coefFicieats -0.519 and -0.723, with p = 0.04 aad 0.002, respectively), Among the 12 wen.@ who underwent surgery within 48 hours after the initial omet of s y m p toms,stahlsinsix~ntsrecoveredmanASr.Agradeof E,inthrcetooneofD,intwotooneofC,ardinooiyone did it remain at a grade of A at tbe censoring tinre (mean follow up 5.24 2 3.25 years), By comparba, of tbe four patients who underwent surgery 48 hours after the initial symptom onset, two died and two remined completely paralytic (ASIA Grade A) after 6- and &.year fobw up, respectively. Furthennore, the median ASIA grades (at 1, 3, 6, and 12 months postoperatively) were s i g n t f d y higher in the 12 patients who underwent surgery within 48 hours than in the four patients in whom the operation was performed after 48 hours (Fig. 5 upper m - w b i t n e y U - ~ t . , p < 0.0051). Of the eight patients in whom a m - pkte neurological dtficits lasted less thiin 12 hours preop eratively, status in five recovered completely within 1 year, in two it recovered to an ASIA grade of D at amsw- ing time, with follow-up periods of 3 and 6 months, respecttvely, and one patient remained completely para- lytic (mean follow up 4.7 1 & 3.5 years). In the eight patients with preoperative complete paralysis lasting more than 12 hours, status in one recovered completely (ASIA Grade E); functimd recovery occunwi in one, though spastic gait remained (ASIA Grade D); ntmfhctional recovery occurred in two in whom muscle strength in half

of the key muscles was less than Grade 3 (ASIA Grade C); and no mzovery was observed in four (ASLA Grade A) iachiding ckath in two cases (mean follow up 5.05 5 3.07 years). Theref=, more rapid recovery and better neu- dugical outcome were observed in s@ly treated patients in whom the duration of deficits was 12 hours or less (Fig. 5 b w r m M M n e y U-test, p < 0.0051).

Two M n t s with p q m t i v e complete deficits and ane with an inccmpb deficit w m dead at the censoring time. The first patient with cervical SSEH died of aspira- ticmpneumonia4~after~operrrtion,andthesec- ond patient, wlsowas receiving ' WY for p a c ~ m a k h$&hm, diod of s- dysfunc- tion3 w ~ a t b G F t h c ~ n . I n b a b c r & s t b e p g t i e n t s did not recover from the complete ne- dysfunc- tion, and the caarses of death weze cons- dated to SSEH. The Ebird cast was a 73-yearold woman in whom SSEH spanned from C2-5 and who pmsaat& with right hemiparesis (her worst muscle sbtjEyph was Grade 2, last- ing for 1 wcck). AAcr evaruation of the cbt, her status recovered to an ASLQ gmde of D by 1 ma&. She died, however, of rcammt,&s 3 years after the SSEH sqery. It was th@t the ckah was not related to SSEH, and the 11cu~olo~caf m v e r y was coded as ASIA Grade D for analysis, Zn addition to the third case, a 74- yew-old woman with popemti= mamp&e deficits did not experience complete m v e a j l 'vely. On MR imaging of the lumbar spine the T f EH dem- ons- an &mnedullary hyper&me signal of the conus mbduh5.s. h l v e hours after developing pcrraly- sis, she mdawent evacuation of dre ch, aad chronic liquelkd dark blood was fwnd -dy. The strength of the L-5 and S-1 key muscles, however, re- mained- 2 at 2 years postoperativeiy, V&cular com- prcnnise presenting as hypuintense spinal d signal may have been the cause of sudden paralysis in the went. Pathological examinations of s p e c b m obi& in 34 patients showed blood dotting. No vascuEar anorndy was found when thc surgical sgdmen was iaiffwIcopically

Six SSEHs in duee p ~ ~ i y qmted cases were described as motor oil-like with fibrotic encapsula- tion, and the cases were considered to be c h m ~ k . ~ The chronic SSEHs involved the ctrvimthwack region in one case, the thoracic region in two, the thoramlumbar spine in one, and the lumbar region in two; the final functional recovery was not related to the acute or CMC character- istics of clotting (Fisher exact test, p = 1).

J. Neurosurg: Spine / Volume 100 /January, 2004 41

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C. C. Liao, et al.

FIG. -3. Sagittal MR images obtained in a 2-year-old boy suffer- - ing from sudden-onset tonic head turning to the left and rapid sen- sorimotor paralysis (ASIA Grade A). A: A T,-weighted image of the cervicothoracic spine revealed an isointense C2-T4 fusiform intraspinal lesion. B: A T,-weighted image demonstrating that the lesion became primarily hyperintense to the spinal cord with foci of hypointensity. After complete neurological deficits persist- ed for 8 hours. the patient undenvent laminectomy and evacuation of the intraspinal lesion. which was proven to be a fresh blood clot. Recovery was complete within 6 months. The SSEH had not re- curred by the 3.5-year follow-up examination.

Discussion

Origins of SSEH

Neither the cause nor pathogenesis of spontaneous epi- dural hematoma is currently clear. Miscellaneous factors such as hypertension.' ingestion of anticoagulant agent^,'^.'^ straining. sneezing, lifting, and spinal vascular anom- alyt'.lq.> 3" have been hypothesized to predispose to this dis- ease. In the present study, the incidences of hypertension in SSEH patients were 34.3% in the emergency department and 20% in the postoperative ward, which are values that are not3ifferent from the prevalence in the adult population in Taiwan (26% in males and 19% in females).32 Therefore, it is presumed that hypertension may coincidently occur with SSEH rather than being a predisposing factor of SSEH. especially given the stressful condition initiated by the symptomatic pain followed by progressive sensorimo- tor paralysis. With regard to the role of anticoagulant ther- apy in the senesis of SSEH. a causal relationship cannot be established because only a small incidence of SSEH was related to the ingestion of warfarin (2.9%) and aspirin (8.4%). and in no case in the present study was there evi-

FIG. 4. Sagittal MR images obtained in a Qmonth-old girl who experienced complete paraplegia 2 hours after an episode of up- per-respiratory tract infection with fever and cough. She was treat- ed for Guillain-Barrk syndrome in a pediatric department for more than I week before cervical MR imaging was performed. The cer- vicothoracic images revealed a hyperintense C7-T7 fusiform lesion on both T,- (A) and TI-weighted (B) images with marked spinal cord compression. The intramedullary hyperintense signal on T,-weighted images represented spinal cord edema and possible infarction. Although she underwent evacuation of the lesion, she remained completely paraplegic with autonomic dysfunction 7.5 years after operation.

dence of bleeding diathesis during the onset. In addition, in millions of people receiving anticoagulant drugs, the worldwide prevalence of SSEH supports no ~ i g ~ c a n t causative fact0r.j

Although the authors of some reports have provided con- vincing radiological or histological evidence that extradu- ral spinal vascular anomaly results in SSEH,9.14.30 no such cases were identified in the present study. It could only be concluded that a vascular anomaly may be an exception- al cause of SSEH. Based on their observation of rapidly evolving signs of SSEH-induced spinal cord compression and based on the fact that the low-pressure epidural veins can be easily tamponaded by normally expanded dural sac dul-ins some surgeries, Beatty and Winston3 assumed that the disruption of small epidural arteries might cause SSEH.

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Spontaneous spinal epidural hematorna . . -.

TABLE 5 . C w postoprcrtivs liruivlogkd ot+quw in 16 patients

- with pgupmRa$ve complete amd 18 pdmts - w&h iAc4unpb neurdogicui d@c&

. - -c --PB

Censoring ASIAOradG Op 1 3 6 92 Tfme

cases wl mmp&e &&it* A 1 6 5 4 4 4 B C 2 2 1 1 D 9 9 5 2 E 3 5 dead 1 2 2 total 16 16 16 is 14t

cases wl inco@wm* A B 3 C 8 D 7 14 11 5 2 E 4 7 13 I S &ad total 18 18 18 18 17s

*Thellacarr~~-qperio8iathe 1 6 p a t a # t t s ~ ~ v e c c # n - plete ckf~its was 483 f: 3.46 ~r#rs.

t m p i a t h m h a d w r ~ b l b w a p o o 1 y s c l a B g t b c ~ t i m e ( o n e f o r 3 a g o n t i r s W o # ~ 6 ~ ~ . ~ ~ . a o u l d w d k indcpc&ently, 7 was admi as ASIA -0, cMB kPaccioaal re- c o v e r y w a s ~ ~ r a e a c h . $ T h e m e p n ~ ~ f l i & l % uddtpopdative incom-

plat dshc$r wm 4.03 t slam. O o w * t ~ ~ ~ a n d ~ ~ w u ~ q &

rrpfor6raoethr. ?barEBvcnd~ywidrioEwm&aBmlamrrptcro- myaadevillEulttiorrbfSBEW.

Gun&ymdHk&ofPbnepacedolr 1 8 ~ , m e n ( 3 9 9 b ) of wRam harbored rn associated hksrtiated bisc, and they h y p d m b d that the M a t e d disc might tear the w e epWd veins of Batson and cause c p % d clotting. An- teddy 1ocakd SSEH, however, is relatively rare in othesr p u w rep~rtsC'~J' and tht prrstnt study. Thfd", tbe hypotfiesis pmposed by G n d y and HeiW cmmt ex- p l a i n t h e ~ e n e s i s i n t h e ~ ~ t y o f S S E H s l o c a t e d ~ y ~ m t t m e s a c . c o o p e t Q a n d M a d d r t l m , e t d . , L d that the onset of S S E H - M sylqmms was llsldly caused by insignificant straining e m , we fortnd this to be so in 19 patients (54%) in our study. Coaper and Madham, et aL, psumed that the pathophysiobgid m e c b i m might involve the transmission of ar tho- racic pnssure to the epiducal venous plexus - &me maneuvers. Because these insigrufieant physiobgkd pro- c e w e s a r e n u m e r o u s i u ~ y l i f c a a d ~ ~ v e development of SSEH in these strahing-aswched &vi- ties is m, a causal relationship amnot bCt mdi&dly es- ~ . Z t i s s U g ~ t h a t t h c ~ o f S S H I may be multifactwial and need further evidence to cknfy its mal origin.

i +

of SSEH TlKl d m i d psentation of SSEH has been well

deScriM%y bmyn and Bosrna? a suddm wset of back or neck pain tfrst watts to c o r m ~ n g demmmes and which is folloaiai*by s i p of nmne root or spinal cord compression. They also d e s c r i i detailed clinical evalu-

Tie dportmn fdlw up

FrG.5. G r a p h S b ~ t h e ~ O f & A s I A * a t l , 3, 6, and 12 months QosBopetrttiv~ly~ Grades w m amvertcd into mmerid equivalents (A, 1; B, 2; and so cm). Death is idcat& by agradeofo. U ~ : ~ i s ~ y b c t t e r i n p a t k n t s unde%oisgj~rg~ywifbiD.48b-theM-onset (Mmn-Wlribwy U-kst, p < &OS). Lower: Better outcomes oocun#iinpatienrsinwtromtbsmaximaldraationofdeficitswas less than 1 2 h o u n s ~ ~ U - a s t , g < 0.05).

ations with symptoms and signs to differentiate miscella- neous lesions simulating SSEH. In view of acute deterio- ration of neurological deficits, pathological entities asso- ciated with spinal cord compression need to be evaluated with rnye1ography, spinal Cf scambg, or MR imaging as soon as pmsiMe.~'4d Although myeiography can demon- st- signs of compwsion-with v h a b t k m of non- spclfic car- blockage or extradutal convex compres- sron-it c-t be used to &tennine the naatrc and the d extension of the l e s i o o . ~ Cornbid with spinal CT scaoning, SSM can k viewed as an intraspina.1 biconvex and h p n s e k s i with the density equivalent to blood. Spinal CT saimhg, however, may be nondiag- -tic in the thoracic spiae wben resolution is poom than i i I in the lumtb6u and cervical spine because of the high con- trast between the hug parcnchyym d vertebral bone? Tkrefixe, in the lllodein era, myelography and CI' scan- ning have been replaced by MB imaging, which provides a noninvasivk and clear view of the precise location of the lesion, its position, size, probable nature, armd the degree of t

cord compression. On T,-weighted imaging, SSEH usual- ly displays isointensc signal to the spinal cord within 24

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C. C. Liao, et al.

hours after symptom onset and hyperintense signal to the cord after 36 Hdtas, et al.,m have suggested that the signal shift from early-stage isohtensity to intermedi- atc-stage hyperintensity on T,-weighted MA images could be pathognomonic for diagnosis of SSEH. Fukui, et al.,12

-- ' however, did not find this to be universally true in their series in which SSEH-related isoinkmity rsisted up to 8 5 days after symptom onset. In addition, S H often caus- es rapid neurological de&eridioa. and the ksion is fie- quently evacuated rapidly after the initial studies. Thus, the MR imaging signal shift demonstrated on the sequen- tial T,-weighted MR images may only be useful in the fol- low-up evaluation of conservatively tnated cases and is not practical in surgery-treattd patients. In all previous reports and this study, T2-weighted imaging demnstrs5ed that almost all SSEH displayed a riwary hypmtens- ity with hypointense foci. Its SAC appearance on T,- weighted images would provide mote characteristic infor- &on for diagnosis, especially when the characteristic s b a l shift on TI-weighted images is lacking during the acute status. Futhamm, its specific T2-weighted signals also aid in the Wmntiation of epidural blood clot from spinal epidural tumor as metastasis or lymphoma that may display the misleading SSEH-related isointense signal on the TI-weighted images.I2 In this study, T,-weighted imag- ing alw revealed increased intensity of the compressed spinal cord, which implied the development of spinal cord edema or ischemia (Fig;. 4). In eight patients with spinal cord edema, functional recovery failed in four (XI%) com- pared with the 10 patients without cord edema in w h o m functional m v e r y OQCW in dl but one (1096). Mag- netic xesomm imaging with T2-weighted uences is potentidy useful for predktmg the newfogi 3 outcome in patients with SSW; this be judged the intra- medullary h m t e n s i t y rtprescnting spinal cold isch- emia. This finding, however, may be due to the small case numbers, because statistic sipficance was not reached (p = 0.12).

Munagemnt and Outcomes of SSEH

In view of the numefous reports, SEH remains arare d unf- clinical entity. Pathobgidy, its c~inid m a n i f e s t a t i o 0 n r n t s f r o m a n ~ i n ~ i n a l ~ - occupying lesion resemblmg the experkmtd model of gradual spinal cumpmsim pdbrmd by TMov.~ He demtmstrated that dogs with ctmphe hindlinib W y s k could only recover aRn clewqwth wbw mt time in- terval was less than 9 hours. R d t s in one of our patients and in dozens nportcd in the littrsauc suggest that favor- able outcoms may be achieved witbout opaatioos when the symptom were mild and could improve spwtaneously after a few hous4J217.1"i341 In cases in wb& the SSEH did nd dLsappear spontaneously in tirm, or in those in which symptoms actively deteriorated spinal cad decom- p n s s i o n s h o u l d b e p e r f ~ a s ~ ~ s p o s s i b l e t o c u r e the spinel dysfunct i021. l~~~ In a review of 333 patients with SSM, Groen end Van Alphtnl5 ooacluded thst surgi- cal evacuation of SSEH within 36 houri; in cases involving complete spinal dysfimction and within 48 hours in those involving incomplete deficits xtslted in favorable out- comes. In the prwent study of 34 surgical cases, preopera- tive status remained the most critical factor to determine

postoperahve neuro1ogical recovery. In patients with in- complete preoperative murological deficits, the recovery rate of nonnal s e w t o r function 1 year postoperative- ly was significantly higher than in those with complete deficits (88.9 and 37.5966, respectively; p < 0.001). Once pnopaatlve mUfOlOgi4 sEstus deteriorated to complete sensorimotor and taattoaomic dy-on (ASIA Grade A), patients who underwent surgery within 48 hours of pain onset experienced si@cantly better recovery than those surgically treated axwe than 48 hours after symptom onset, as d e m m d at 1,3,6, and 12 months postoperatively (p < 0.005). SimiZariy, better results were obsemed in pa- tients who underwent surgery when the duration of neuro- logical dysfunction was less than 12 hours compared with more than 12 hours (p < 0.005).

In our analysis, age, sex, location or extension of the SSEH, and the rate of evolutiun of symptoms and signs of SSEH-induced spinal cord compmsbn were not correlat- ed with the postoperative neudogiicat uutcomes. It is diffi- cult, however, to establish the ahsolute time limit to ensure comple$e n t u m ~ c a l recuvery because some neurological recoveris occurred &&rably longer after surgical in- tenention. Although the actual fbce aod time interval re- quired to produce s SSm-indnrced spinal cord compression may n z - we suggest that the SSEH can be viewed as a chiad mode1 that gradually causes splaal cad compssh, as in the dog model pro- posed by Tarlov," and fb&mmm that it shoarM be treated by aggresivc and eme%eacy thmqmsive surgery at o n c e i f c o m p m ~ ~ ~ s ~ .

Spontaneous spinal epidural h e m a - is a rare but dis- abling surgical disorder. Its origin is scarcely correlated with Bypatension, anbwagdation therapy, and extradu- ral spinal vwaalar a ~ 8 o d y . Althougb this could not be proven based on c m t evidence, shifting of intrathoracic or intraabhmhl prcswe during saane physidogkal ex- erck may ruptuzla tht e p h d vemus plexus and result inepidmd- Suddenonsetof~korneckpain followed by rapid se- paralysis due to hema- tom-induced c a n p s i o n of the spinal cord or nerve roots is a welldocumented and mid presentation. Prompt MR imaging examination can debxk the in- traspiBal lesioos in patients with acute spural cord com- pmsion syndmme. FwthennOre, T,-we@ted MR imag- ing provides additional infomatb to determine the nature of SSEH. Surgcry k a safe, effective, and indis- pensable method of treating the disease, with a related complication rate of 2.9%. Although the SSEH-related mortality rate was 5.7% in the present series, there was no operation-related fatality. One hundred percent functional recovery a d 88.9% complete nemIogical recovery were whieved in patients with pmperative incomplete neuro- logical deficits. The result is sipdkantly better than that in patients with preqxmtive complete neurological defi- cits ( 0 0 ~ nemmogical recovery rate 37.5%; p < 0.001). In patients wim complete (ASIA Grade A) defi- cits, good neumIogical recovery can also be achieved after timely surgery when the interval between the initial ictus (mostly pain) and surgery is less than 48 hours and when

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Spontaneous spinal epidural hematoma -.

the durations of tlme compltte nemlogml deficits is less than 12 burs.

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hamus@% d v e d April 4,2003. Accepted in f'iual fibna August 25,2003.

kr: Shih-T~~ng Lce, M.D., -t o f ~ * ~ G u P g h a c m o l . I a i ~ t a l , 5 , F u - ~ S ~ 333, Kweishaa, Taoyuan, Taiwan. email: [email protected]. 0rg.w.

I. Ncurosurg: Spine / Volwne I W / J&, 2004