CASE REPORT
Ch r on ic pe lvic pa in : t h e r ole of t h e n u t cr a ck e r syn dr om e
M a r ce lo Fe r r e ir aI, Lu iz La n z iot t iI, Gia fa r Abu h a dbaI, M a r ce lo M on t e ir oI, Lu is Ca pot or t oI, José Lu iz Spica cciI I
IServiço I nt egr ado de Técnicas Endovascular es ( SI TE) , Rio de Janeir o, RJ, Br azil. I IHospit al Sam ar it ano, Rio de Janeir o, RJ, Brazil.
Correspondence
J Vasc Bras. 2008; 7( 1) : 76- 9.
ABSTRACT
Chr onic pelvic pain is a pr oblem at t he sam e t im e com m on and under diagnosed in w om en. Som e lit erat ure report s show an incidence of up t o 15% in w om en aged bet w een 18- 50 years, w it h im pact over qualit y of life and econom y. Am ong t he causes of chronic pelvic pain, pelvic congest ion syndr om e st ands out , charact erized by pain, dysur ia, hem at ur ia, dysm enor r hea, dyspareunia and vulvar congest ion, oft en accom panied by vulvar var ices, descr ibed in 1949 by Taylor. We herein report a case of a pat ient w it h chronic pelvic pain in w hom w e diagnosed a nut cracker syndr om e, charact erized by st enosis of t he left r enal vein bet w een t he superior m esent er ic ar t er y and t he aort a, w it h consequent st at us of left gonadal plexus hypert ension, pelvic varices and sym pt om s of pelvic congest ion. The t r eat m ent w as var icose pelvic veins em bolizat ion, t hr ough a m inim ally invasive endovascular approach, w it h im m ediat e t echnical and clinical success in less t han 24 hour s.
Ke yw or ds:Pelvic pain, var icose veins, r enal veins, em bolizat ion, angioplast y.
RESUM O
esquerda ent re a aort a e a art éria m esent ér ica superior, com conseqüent e quadr o de hiper t ensão do plexo gonadal esquer do, varizes pélvicas e sint om as de congest ão pélvica. O t rat am ent o realizado const ou de em bolização das varizes pélvicas, por m ét odo m inim am ent e invasivo endovascular, com sucesso t écnico e r esolução dos sint om as em m enos de 24 h.
Pa la vr a s- ch a ve : Dor pélvica, var izes, veias r enais, em bolização, angioplast ia.
I n t r odu ct ion
Chr onic pelvic pain is a com m on and under diagnosed pr oblem in w om en. I n a st udy including w om en aged bet w een 18- 50 years, Mat hias et al. found a 15% prevalence, w it h m aj or
consequences on qualit y of life and econom y.1 Am ong t he causes of chr onic pelvic pain, pelv ic congest ion syndr om e st ands out , w it h clinical st at us charact erized by var ied degr ees of pain, dysuria, dysm enor r hea, dyspareunia and vulvar congest ion, oft en accom panied by vulvar var ices,2 -6 described in 1949 by Taylor .2
Laborat ory t est s for t hose cases frequent ly show signs of m icrohem at uria, usually associat ed w it h t he nut cracker syndrom e, an anat om ical var iat ion in w hich t he superior m esent er ic art ery ( SMA) and t he aort a per for m a clam ping of t he left r enal vein, w it h consequent r eflux in t he pr oxim al por t ion of t hat vein and of t he left ovar ian vein. The nut cr acker syndr om e usually affect s w om en aged bet w een 20- 40 years, especially m ult ipar ous w om en, and venous reflux causes var icose veins of t he deep and superficial pelvic venous plexus, responsible for a clinical st at us t ypical of left flank and chronic abdom inal pain, besides m icr ohem at ur ia.3 , 4 , 7 - 1 1 I n m en, t his syndr om e can be
m anifest ed sim ilar ly, being one of t he descr ibed causes of var icocele.
Ca se r e por t
A 35- year - old pat ient , m ult ipar ous, businessper son, sought for clinical care present ing abdom inal, pelvic and left flank pain for about 2 years, w it h daily use of incr easing doses of analgesics,
including opioids, w it h no t her apeut ic success, despit e a long invest igat ional hist or y com bined w it h several im aging exam inat ions and laborat ory t est s.
The pat ient w as r efer r ed t o t he vascular and endovascular surgery ser vice t o evaluat e possibilit y of a m inim ally invasive t reat m ent . She w as t hen subm it t ed t o venogr aphy, show ing clam ping of t he left r enal vein, w it h significant incr ease in it s pr oxim al diam et er , besides ext ensive var ices of t he deep pelvic venous plexus, w it h invert ed venous r eflux in t he left ovarian vein, w hich also had a diam et er about t w o t o t hree t im es larger t han expect ed (Figure 2) .
Tw o endovascular t herapeut ic alt er nat ives w er e init ially considered: use of st ent in t he left r enal vein and em bolizat ion of pelvic var ices, bot h being procedures r epor t ed in t he int er nat ional lit erat ure t o r epair t hat clinical st at us.3 , 1 2 - 1 8
Clinical course was satisfactory, with complete regression of pain 24 hours after the procedure. Doppler ultrasound in the immediate postoperative period showed absence of flow in embolized veins, and the patient was discharged 72 hours after the procedure, with suspension of analgesic drugs.
D iscu ssion
Prevalence of the nutcracker syndrome is relatively much higher than its diagnosis, probably
because the presence of characteristic anatomic changes does not always cause symptoms.4
With regard to cases in which it is confirmedly responsible for symptoms of pelvic venous
congestion, lack of clinical reports in the current gynecological and obstetric literature shows that venous congestion is not considered in clinical investigation with adequate frequency. In a study including 66 patients between 1992 and 2000, d’Archambeau et al. reported an 83% incidence of
the nutcracker syndrome in patients referred due to symptoms of pelvic venous congestion.9
Diagnostic of pelvic venous congestion should be considered among causes of chronic pelvic pain, especially after exclusion of other more common causes, such as pelvic inflammatory disease, endometriosis, interstitial cystitis, pelvic tumors or intestinal inflammatory disease. As to complementary examinations, ultrasound associated with vascular Doppler can be used as a scr eeningexamination, as long as a proper intestinal preparation is performed and the examiner is experienced enough to identify affected vascular structures. Both magnetic nuclear resonance and tomography can be used, with high sensitivity and specificity, despite being considered invasive.
In d’Archambeau's series,9treatment of choice was the endovascular, through embolization of the
left ovarian vein and underlying pelvic varicosities, with initial clinical success rate of 86% and reduction of 73% in long-term clinical complaints (mean 43.4 months).
Treatment of pelvic venous congestion and the nutcracker syndrome can be through drug treatment, using estrogens and anti-inflammatory agents, but it has poor therapeutic response.
Our choice is for t he endovascular m et hod, w hich w e believe brings excellent benefit s and is t he first - choice m et hod in current lit erat ure. I n addit ion, it is m inim ally invasive, as dem onst r at ed in t his case, w hich had excellent clinical cour se aft er a 6- m ont h post operat ive follow- up.
There is a need of publishing m ore inform at ion on t he nut cr acker syndr om e and on it s t reat m ent in varied specialt ies involved, especially ult r asound, gynecology and urology.
Re fe r e n ce s
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prevalence, healt h- relat ed qualit y of life, and econom ic correlat es. Obst et Gynecol. 1996; 87: 321- 7.
2. Taylor HC Jr . Vascular congest ion and hyper em ia: t he effect on st r uct ur e and funct ion in t he
fem ale r epr oduct ive syst em .Am J Obst et Gynecol. 1949; 57: 211- 30.
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2005; 42: 275- 80.
Cor r e spon de n ce :
Marcelo Fer r eir a
Rua Siqueira Cam pos, 59/ 203, Copacabana CEP 22031- 070 - Rio de Janeir o, RJ, Brazil Tel.: ( 21) 3816.0160
Em ail: m m vf@uol.com .br
