CASE REPORT
D ia gn osis a n d t r e a t m e n t of pe r sist e n t scia t ic a r t e r y a n e u r ysm : ca se
r e por t a n d r e vie w of t h e lit e r a t u r e
M a r co An t on io Pr a do N u n e sI, Robe r t o M a u r ício Fe r r e ir a Ribe ir oI I, José Ade r va l Ar a gã oI I I, Fr a n cisco Pr a do Re isI V, V e r a Lú cia Cor r e a Fe it osaV
IAssist ant pr ofessor , Univer sidade Tir adent es ( UNI T) , Ar acaj u, SE, Br azil.
I IAngiologist , Hospit al de Urgência de Sergipe João Alves Filho, Ar acaj u, SE, Brazil.
I I IAssist ant pr ofessor , UNI T, Aracaj u, SE, Br azil. Assist ant professor, Univer sidade Feder al de
Sergipe ( UFS) , Aracaj u, SE, Brazil.
I VProfessor, UNI T, Ar acaj u, SE, Br azil.
VAssist ant pr ofessor , UFS, Aracaj u, SE, Brazil.
Correspondence
J Vasc Bras. 2008; 7( 1) : 66- 71.
ABSTRACT
Persist ent sciat ic art ery is a r ar e anat om ical var iat ion w hose clinical cour se is pot ent ially serious, since diagnosis is perform ed w hen clinical com plicat ions ar e alr eady present . Such com plicat ions are a consequence of t he fact t hat sciat ic art ery w alls have a t endency t o at her oscler ot ic
degener at ion w it h aneur ysm al form at ion and m ay evolve t o occlusive t hr om bosis or t o
t hr om boem bolism . This art icle r epor t s t he case of a fem ale pat ient w it h com plaint of int ense pain secondary t o ext r insic com pr ession of t he sciat ic ner ve by aneurysm al dilat at ion of t he r ight low er m em ber and hyperpulsat ile ipsilat eral but t ock. Angiogr aphic exam inat ion w as car r ied t hrough, show ing com plet e per sist ent sciat ic art ery t o t he r ight low er m em ber w it h an aneur ysm . The pat ient w as subm it t ed t o a surgical pr ocedur e using a t r ansglut eal appr oach and a t er m
ino-t erm inal Dacron gr afino-t w as inino-t er posed beino-t w een ino-t he pr oxim al and disino-t al necks of ino-t he aneur ysm . The t echnical aspect s and review of lit er at ur e on diagnosis and t her apeut ic approach of t his anat om ical variat ion are discussed in t his w ork.
Ke yw or ds:Persist ent sciat ic art ery, sciat ic art ery aneur ysm , sur gical cor r ect ion, per ipher al ischem ia.
A persist ência da ar t ér ia isquiát ica é um a var iação anat ôm ica rara cuj o cur so clínico é
pot encialm ent e gr ave, pois o diagnóst ico ocorre quando j á est ão pr esent es com plicações clínicas. Essas com plicações advêm do fat o de a par ede da ar t ér ia isquiát ica t er um a t endência à
degener ação at er om at osa com for m ação aneur ism át ica, podendo evoluir par a um a oclusão ou para um t r om boem bolism o. Est e ar t igo relat a o caso de um pacient e do sexo fem inino, com queix as de dor int ensa secundária a com pressão ext rínseca do ner vo isquiát ico pela dilat ação aneur ism át ica no m em bro inferior direit o e pulsat ilidade na nádega ipsilat eral, apr esent ando um a m assa hiperpulsát il nessa localização. Foi realizado exam e angiográfico, que revelou persist ência da art éria isquiát ica à direit a, do t ipo com plet o e com aneurism a. A pacient e foi subm et ida a pr ocedim ent o cirúrgico, sendo ut ilizada um a abordagem t r ansglút ea, e um a pr ót ese de dácron t er m inot er m inal foi int erpost a ent r e os colos proxim al e dist al do aneur ism a. Os aspect os t écnicos e r evisão da lit erat ura sobre o diagnóst ico e a t er apêut ica dessa var iação anat ôm ica são discut idos nest e t rabalho.
Pa la vr a s- ch a ve : Per sist ência da ar t ér ia isquiát ica, aneur ism a da art éria isquiát ica, t er apêut ica cirúrgica, isquem ia per ifér ica.
I n t r odu ct ion
Sciat ic art ery is an axial art ery em br yologically r esponsible for t he blood supply of low er lim bs. Under nor m al condit ions, around t he t hir d m ont h of em br yonic life, it regresses t o form t he proxim al segm ent of t he infer ior glut eal ar t er y. This occurs aft er t he developm ent of t he fem or al art ery from t he ext er nal iliac art ery.1
I n case of per sist ent sciat ic ar t er y, t he super ficial fem or al art ery under goes a pr ocess of at resia in t he dist al t high, and is usually int errupt ed at t he adduct or hiat us level, ent er ing t he leg as post er ior t ibial ar t er y. The fem or al art ery is occasionally ver y short and ends as t he com m on deep circum flex t runk.2 Persist ent sciat ic art ery is a r ar e anat om ic var iat ion and t ends t o for m at ion of aneur ysm ,
at herosclerosis or dist al em bolizat ion.3
I kezaw a et al.4 perform ed a r eview of t he available lit erat ure and r epor t ed occur r ence of 168
cases. Persist ent sciat ic art ery is pot ent ially sever e, since it has a pr im it ive vascular t r unk t hat plays t he r ole of t he m ain supply vessel for t he w hole low er lim b.2 On t he ot her hand, diagnosis usually occurs w hen clinical com plicat ions are alr eady present . Such com plicat ions result fr om t he fact t hat sciat ic art ery w all t ends t o at her om at ous degener at ion w it h aneurysm al for m at ion, and m ay pr ogr ess t o occlusion or t hr om boem bolism .3
Early det ect ion of per sist ent sciat ic art ery allow s prophylact ic surgical t herapy, pr event ing pot ent ial and severe com plicat ions, w hich can com pr om ise low er lim b cir culat ion.2 Tr eat m ent of lesions
result ing fr om per sist ent sciat ic art ery is ver y har d, due t o it s rarit y and lack of exper ience by healt h pr ofessionals, w ho are usually unaw ar e of it s course and have difficult y choosing t he best t reat m ent for t hose lesions. Therefore, it is im port ant t o per for m fur t her st udies on t his issue, aim ing t o m ake early diagnosis feasible and provide a bet t er follow- up of such com plicat ions.
A 55- year - old fem ale pat ient , com plaining of int ense secondary pain at int rinsic com pr ession of t he sciat ic ner ve by aneurysm al dilat at ion in t he r ight low er lim b and pulsat ilit y in t he ipsilat eral
but t ock w as at t ended at t he hem odynam ic sect or t o under go low er lim b ar t er iogr aphy. On physical exam inat ion, t here w as pr esence of a hyperpulsat ile m ass in t he r ight but t ock. On pulse
assessm ent , r ight poplit eal art ery pulse w as nor m al, fem or al art ery pulse w as very w eak and t ibial post er ior and pedal pulses w er e absent . On t he left , all pulses w er e pr esent and nor m al.
An angiographic exam inat ion w as perform ed t hr ough punct ure of t he left com m on fem or al art ery w it h select ive cat het erizat ion of t he r ight com m on iliac art ery. Ther e w as pr esence of hypoplasia of t he r ight fem or al art ery, w hose diam et er w as reduced, going m edially and ending at t he t high dist al level, from w her e som e m uscle br anches em er ged. Ther e w as also dilat at ion of t he r ight int ernal iliac art ery (Figure 1) and post er ior cir culat ion of t he r ight low er lim b supplied by t he sciat ic art ery. I t had focal fusifor m aneur ysm al dilat at ion in t he r ight glut eal r egion (Figure 2) , st enosis in it s dist al neck and follow ed a descending and post er ior rout e along t he t high (Figure 3) unt il r eaching t he adduct or canal, w her e it cont inued as poplit eal art ery. Aft er poplit eal art ery filling, leg ar t er ies could not be visualized. Ther e w as no per sist ent sciat ic art ery in t he left low er lim b.
Due t o risk of dist al em bolizat ion and int ense pain in t he r ight low er lim b, t he pat ient w as subm it t ed t o a sur gical pr ocedur e, using a t r ansglut eal appr oach t o expose t he sciat ic art ery aneurysm , being car eful t o ident ify and avoid sciat ic ner ve lesion. Aft er dissect ion and clam ping of dist al and proxim al necks, t he aneur ysm w as open and a large t hr om bus w as r em oved fr om t he aneurysm al sac. Ther e w as no evidence of collat eral vessels inside t he aneur ysm . A
t er m inot er m inal Dacron graft w as int er posed bet w een t he proxim al and dist al necks, and t he aneurysm al sac w as closed over t he graft aft er par t if w as r em oved. One m ont h aft er t he surgery, t he pat ient r epor t ed pain relief and w as subm it t ed t o art erial color flow Doppler ult r asound,
D iscu ssion
I ncidence of per sist ent sciat ic art ery based on angiogr aphic exam inat ions is est im at ed t o be in 0.05% ,2 and w hen assessed by angiogr aphic t om ogr aphy in pat ient s w it h clinical st at us of acut e
and/ or chronic ischem ia, such incidence r eaches appr oxim at ely 1.63% .5 So far t here have been no st udies t o est im at e it s incidence in Br azil. St udies carried out by Br azilian aut hor s r epor t ed cases including age and gender . Thus, Dias et al.,6 Oliveir a et al.,7 Araúj o et al.8 and Bez et al.9
described a var iat ion bet w een 58- 74 years, w it h m ean of 64.25 years, t hree cases being in w om en6 - 8 and only one in t he m ale gender .9 Age r anged bet w een 42- 82 for Maldini et al.,1 0 w it h
m ean of 62 years. The pr esent st udy included a 55- year - old fem ale pat ient .
Bilat eralit y in per sist ent sciat ic ar t er y, accor ding t o Mayschak & Flye,2 is pr esent in 50% of cases.
Araúj o et al.8 described one bilat er al case. Dias et al.6 and Bez et al.9 found per sist ence in t he r ight
There are m any t ypes of per sist ent sciat ic art ery pr esent at ion. I t is called com plet e w hen t he sciat ic art ery ser ves as dir ect com m unicat ion bet w een t he int ernal iliac art ery and t he poplit eal art ery. I n such cases, t he hypoplast ic super ficial fem or al art ery usually com m unicat es w it h t he poplit eal art ery t hr ough a variable net w or k of collat eral vessels.1 1 Mayschak & Flye2 r epor t ed t hat m ost cases found in t heir st udy included com plet e sciat ic ar t er y. Our st udy corroborat es t he report s per for m ed by Dias et al.,6 Oliveir a et al.,7 Araúj o et al.8 and Bez et al.,9 and w e found t he
com plet e t ype.
Present at ion is called incom plet e w hen t her e is part ial involut ion of t he sciat ic art ery. I t becom es hypoplast ic and com m unicat es w it h t he poplit eal art ery t hr ough a net w or k of collat er al vessels, besides present ing a nor m al fem or al syst em .1 0
As t o incidence of sciat ic art ery aneur ysm , according t o Mayschak & Flye2 it cor r esponds t o 15% of cases and usually occurs under t he glut eus m axim us m uscle at t he great er t r ochant er level. To Maldini et al.,1 0 incidence m ay vary bet w een 15- 46% . I n t he sur veyed lit erat ure, t here w er e 87
report s of sciat ic art ery aneur ysm unt il 2002. The basic et iology of aneur ysm al dilat at ion in persist ent sciat ic art ery has not been w ell est ablished.1 0 Com par ed t o t he iliac- fem or al segm ent , persist ent sciat ic art ery t ends t o a lar ger at her om at ous degener at ion and it s post er ior course t o glut eal and t high r egions, w here it is subj ect t o r ecur r ent t r aum as, favoring for m at ion of aneur ysm , rupt ure, t hr om bosis and at herom a em bolizat ion. Dias et al.,6 Oliveir a et al.7 and Araúj o et al.8
report ed t hree cases w it h pr esence of aneur ysm . I n t he last st udy, a case of bilat er al aneurysm w as described, st ressing t hat t here w as r upt ur e w it h hem at om a in t he r ight side, besides sciat ic art ery occlusion and acut e ischem ia of t he affect ed lim b.
Diagnosis of per sist ent sciat ic art ery dem ands a high level of suspicion by t he physician, besides being dependent on t he pat ient 's clinical pr esent at ion and physical exam inat ion. I t should also be st ressed t he im por t ance of anat om ic configur at ion of t he per sist ent sciat ic art ery and w het her or not it present s associat ed aneur ysm . Clinical signs and sym pt om s ar e usually det er m ined by com plicat ions. I t is ver y im port ant t o exclude hist or y of r ecent t r aum a t o avoid false diagnosis of aneurysm .2 I n m or e t han 40% of cases, per sist ence is asym pt om at ic and diagnosed t hr ough
accident al findings, unr elat ed t o for m at ion of aneur ysm or ot her com plicat ions.1 0
To Wilson et al.,1 1 absence of fem or al pulses and pr esence of dist al pulses r epr esent a high level of
suspicion of per sist ent sciat ic art ery. I n hypoplasia or absence of fem or al art ery, t he pat ient has palpable poplit eal and pedal pulses, despit e absence or slight reduct ion in fem or al pulse am plit ude. Such present at ion, called Cow ie's signal, is st rongly suggest ive of com plet e per sist ent sciat ic art ery.
Pain in t he glut eal r egion is consider ed a nonspecific sy m pt om .1 1 I t is adm it t ed t hat it occurs due t o pr oxim it y of sciat ic art ery and aneurysm w it h sciat ic nerve, w hich can also cause sensit ive or m ot or changes in t he affect ed lim b. Mayschak & Flye2 and Maldini et al.1 0 adm it t hat pr esence of a
painful m ass in t he glut eal r egion m ay occur. Accor ding t o Mayschak & Flye,2 t hat sam e m ass is
pulsat ile and could r epr esent aneur ysm al dilat at ion of t he art ery. I kezaw a et al.4 found glut eal m ass in 25.7% of cases t hey invest igat ed.
The m ost frequent clinical finding in per sist ent sciat ic art ery is low er lim b ischem ia. To Maldini et al.1 0 it can occur in 63% of cases, and in 25% of t hem it can be m anifest ed as cr it ical ischem ia. I kesaw a et al.4 r epor t ed t hat 31.1% of pat ient s w it h sym pt om at ic per sist ent sciat ic art ery had ischem ia. Wilson et al.1 1 r epor t ed t hat rat e of aneur ysm for m at ion in cases of per sist ent sciat ic
art ery is high. For it s diagnosis, t he m ost w idely used inst rum ent is digit al subt r act ion angiography.5 I t is st ill essent ial t o det er m ine vascularizat ion pat t er n and pr ovide necessary
for a proper analysis of angiogr aphic findings.
Color - flow Doppler ult rasound is anot her diagnost ic resource t hat helps det ect per sist ent sciat ic art ery. How ever , com put ed t om ography and angiogr aphy st and out in diagnost ic confir m at ion, since t hey show var iat ion in anat om ic configurat ion of sciat ic and fem or al art erial syst em s.1 3
Angiographic t om ogr aphy allow s det ect ion of occluded sciat ic art ery, w hich cannot be visualized by digit al angiogr aphy, besides pr oviding assessm ent of differ ent com plicat ions, such as aneurysm s, degree of int r am ur al t hrom bosis, vascular anat om y, art ery cour se and it s r elat ion w it h adj acent st ruct ures and possible associat ed venous anom alies.
I n t he pr esent st udy, angiogr aphic exam inat ion w as per for m ed, as w ell as in t he st udied by Oliveira et al.7 and Araúj o et al.8 Dias et al.6 added color- flow Doppler ult rasound t o angiogr aphy, and Bez et al.9 added angiographic t om ogr aphy. Jung et al.5 concluded t hat angiogr aphic
t om ography could be used as t he only inst r um ent for pret herapeut ic assessm ent of any
t hrom boem bolic com plicat ion or at her oscler ot ic change. Angiogr aphy t hr ough m agnet ic r esonance can be consider ed as a good diagnost ic inst r um ent . How ever , high cost and t echnical difficult y rest rict it s use.1 2
I n case of per sist ent sciat ic ar t er y, choice of t herapeut ic conduct is dependent on art ery anat om y and occurrence of com plicat ions.1 3 To Jung et al.5 t he asym pt om at ic for m of t hat per sist ence,
alt hough it does not r equir e sur gical pr ocedur e, should be m onit or ed due t o a high risk of t hrom boem bolic com plicat ions. Early det ect ion, t her efor e, allow s a m or e im m ediat e sur gical t her apy t o prevent pot ent ial and ser ious com plicat ions.2
I n t he pr esence of ischem ic com plicat ions, indicat ion of a sur gical t reat m ent is absolut e, especially due t o risk of chronic ischem ia result ing from dist al t hr om boem bolism or fr om aneur ysm
t hr om bosis.1 4 I n com plet e aneur ysm exclusion, suggest ion is for som e t ype of revascularizat ion of
t he super ficial fem or al art ery hypoplast ic syst em .1 1 I n such cases, t her e are report s of som e t ypes
of sur gical pr ocedur es, such as aneur ysm r esect ion and dir ect anast om osis bet w een t w o vessel ext r em it ies, gr aft int er posit ion, art ery exclusion w it h pr oxim al and dist al ligat ion, iliac or
fem oropoplit eal bypassand endoaneur ysm or r aphy.1 4 I n our case, t he pat ient w as subm it t ed t o a surgical pr ocedur e in w hich t he aneurysm w as excised w it h gr aft int er posit ion and r em oval of a large t hr om bus, follow ed by int er posit ion of a Dacron graft w it h t er m inot er m inal anast om osis. Based on t he w ar ning by I shida et al.,1 5 dur ing aneurysm resect ion, w e w er e careful enough not t o cause any lesion t o t he sciat ic nerve.
Surgical pr ocedur es, com bined w it h endovascular t echniques for aneurysm occlusion, according t o Loh,1 6 avoid need and r isks of surgery on t he sciat ic ar t er y. Sim ple art erial em bolizat ion is
r ecom m ended by Song et al.3 for t he t reat m ent of com plet e and incom plet e sciat ic art ery
aneurysm s, w hen t here is a good net w or k of collat er al vessels. Sogaro et al.1 4 suggest choice of
em bolizat ion using disposable balloons in cases of incom plet e t ype. I n com plet e t ypes, a cover ed st ent could be im plant ed by endovascular t echnique.
Most post sur gical com plicat ions occur in pat ient s w it h com plet e per sist ent sciat ic art ery. These pat ient s do not gener ally have a good net w or k of collat eral vessels.3 Dias et al.6 r epor t ed t hat , in
one case, low er lim b am put at ion w as per for m ed. Accor ding t o Jung et al.,5 t hose sit uat ions occur
due t o delay in sear ch for m edical t r eat m ent . I n our case, t he pat ient pr ogr essed unevent fully and, 1 m ont h aft er t he sur ger y, ar t er ial Doppler ult r asound show ed good gr aft pat ency.
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