Sacral Neuromodulation: Foray into Chronic Pelvic Pain in End Stage Endometriosis

Case Report Sacral Neuromodulation: Foray into Chronic Pelvic Pain in End Stage Endometriosis Maija Lavonius, Pia Suvitie, Pirita Varpe, and Heikki Huhtinen Department of Digestive Surgery and Department of Gynecology and Obstetrics, Turku University Hospital, Turku, Finland CorrespondenceshouldbeaddressedtoMaijaLavonius;[email protected] Received 28 August 2016; Revised 11 January 2017; Accepted 19 February 2017; Published 6 March 2017 A cademicEditor:DominicB.Fee Copyright © 2017 MaijaLavoniusetal.ThisisanopenaccessarticledistributedundertheCreativeCommonsAttributionLicense, whichpermitsunrestricteduse,distribution,andreproductio ninanymedium,providedtheoriginalworkisproperlycited. Excisionofallendometrioticlesionsisthemethodofchoiceinthetreatmentofsevereendometriosisresistanttomedicaltherapy. Theinfiltratingnatureofthediseaseaswellasextensivesurgerymay,however,causechronicpainthatcannotberelievedbyeither surgeryorhormonaltreatment.Asapilottreatment,wetestedtheeffectofsacralneuromodulation(SNM)forfourendometriosis patients suffering chronic pelvic pain and pelvic organ dysfunction after radical surgical treatment. Three out of four patients reported improvement in their symptoms during the neuromodulation testing period and a permanent pulse generator was installed. After 2.5 years, all three patients report better quality of life and want to continue with SNM. 1. Introduction Sacral neuromodulation (SNM) involves electrical modula- tion of a sacral nerve root by means of an electrode and a pulse generator. The conventional indications for SNM are urinary retention, urinary incontinence, anal incontinence, and constipation [1]. Several studies have also demonstrated promisingresultsofSNMinchronicpelvicpain[2],although its mechanism of action remains ill defined [3]. One study hasevalua tedtheeffectofSNMonvoidingdysfunctionafter surgeryfordeepinfiltrating(DIE)endometriosis[4].Toour knowledge,noreportsofSNMforthetreatmentofpelvicpain afterradicalsurgeryofendometriosishavebeenpublished. Endometriosisisanestrogen-dependentchronicinflam- matory disease defined by the presence of functional endo- m e t r i a lt i s s u eo u t s i d et h eu t e r i n ec a v i t y .A p p r o x i m a t e l y4 – 10%ofwomenareaffectedduringtheirfertileyearssuffering from a variety of pelvic pain symptoms and subfertility [5]. Endometriosis significantly reduces health-related quality of life (HRQoL) mostly due to pain symptoms [6]. Pain can be alleviated with hormonal medication and nonsteroidal anti- inflammatory drugs (NSAID) [7]. When medical therapy fails, complete surgical excision of the endometriotic lesions is the method of choice. In severe DIE, a multidisciplinary a p p r oachcanbenecessary . Endometriosis can cause pain in many different mech- anisms [8]. Irritation of pelvic nerves due to inflammation causesnociceptivepain.Inaddition,DIEcaninfiltratepelvic nerves causing pain, and de novo nerve growth is also detected in ectopic endometrial tissue. During surgery, all efforts are made to preserve the pelvic nerves. However, in severe cases, nerve-sparing technique is not possible if the goal is to remove all endometriosis. In such cases, a compromise is made between radicalness of surgery and preservation of pelvic organ function. The disease itself as well as extensive surgery can lead to chronic neuropathic pain.Insuchcases,littleoptionsareavailabletoeasethepain. 2. Case Presentation Common to all our four patients was a history of sur- gically treated deep infiltrating endometriosis in lateral pelvicsidewallsaffectingureters,sacrouterineligaments,and pelvic nerves. All women had undergone hysterectomy and bilateral salpingo-oophorectomy combined with excision of all macroscopic endometriosis. Thus, adenomyosis as the source of pain was excluded. No recurrent endometriosis wassuspectedingynecologicalexaminationandtransvaginal ultrasound, and the chronic noncyclical pelvic pain was life interferingandresistanttohormonalandmedicaltreatment. Hindawi Case Reports in Neurological Medicine Volume 2017, Article ID 2197831, 4 pages https://doi.org/10.1155/2017/2197831 2 CaseReportsinNeurologicalMedicine Table1:Surgicalhistoryofthefourwomenintheneuromodulationpilotstudy.Operationswereperformedbylaparotomyiflaparoscopyis notmentionedinthetext. Patient Age (years) year Operation Indication Postoperative complications 1 G2P2 1999 Laparoscopy Endometriosis None 2000 Laparoscopicleftsalpingo-oophorectomy Endometriosis None 2000 Laparoscopic right salpingo-oophorectomy, supracervicalhysterectomy Endometriosis None 2009 Adhesiolysisandextirpationoftheuterinecervix Pelvicpain None 2011 Anteriorresection,appendicectomy,and adhesiolysis Pelvicpain(leftside) None 43 2014 Neuromodulationtestperiodandapermanentpulse generatorinstallation(rightS4) Pelvicpain None 2 G0P0 2002 Leftsalpingo-oophorectomyandadhesiolysis Endometriosis None 2006 Anteriorresectionandileostomy Sigmoidperforation Rectalanastomosis stricture 2007 Reanteriorresectionandileostomyclosure None 2009 Adhesiolysis,ileumresection,andsupracervical hysterectomy Postoperativeadhesions, bowelobstruction None 2013 Re-reanteriorresection,resectionofleftureter, extirpationoftheuterinecervix,andileostomy Endometriosis(main symptompain)andanal incontinenceand defecationdifficulties Ureteralanastomosis leakagetreatedby pyelostomyanddrainage 2013 Ileostomyclosure None 50 2014 Neuromodulationtestperiodandapermanentpulse generatorinstallation(leftS4) Pelvicpainanddefecation difficulties None 3 G2P2 1991 Laparoscopy Endometriosis None 1995 Laparoscopy Endometriosis None 2003 Laparoscopicexcisionofsacrouterineligamentsand electrocoagulationofperitonealendometriosis Endometriosis None 2005 Laparoscopicelectrocoagulationofperitoneal endometriosisandadhesiolysis Endometriosis None 2008 Hysterectomyandexcisionofperitoneal endometriosis Endometriosis None 2010 Laparoscopicadhesiolysisandleft salpingo-oophorectomy Postoperativeadhesions, pain,leftovariancyst None 2010 Vaginal resection, extirpation of left periureteral endometriosis Endometriosis, hydronephrosis Laparotomywound infection 42 2014 Neuromodulationtestingperiodandapermanent pulsegeneratorinstallation(rightS4) Pelvicpainanddefecation difficulties None 4 G1P0 2011 Hysterectomy andbilateral salpingectomy Endometriosis None 2012 Ileumresection,appendicectomy,bilateral oophorectomy,vaginalresection,andresectionof leftsacrouterineligament Endometriosis,pelvicpain None 35 2014 Neuromodulationtestingperiod(leftS3) Pelvicpain None Note.G:gra vidi ty ,P:pari ty . Detailed surgical history is presented in Table 1. All women were fully informed of the experimental nature of the SNM treatmentforthisindication.Noethicsapprovalwasapplied because of acknowledged use of SNM for other indications with pelvic disorders. SNM was offered to one more patient butshedeclinedbecauseshefearedtheprocedure. The SNM procedure with InterStim II/_0054system was performed in two stages. In the first stage, a permanent lead was implanted under local anesthesia with fluoroscopic control into S3 or S4 sacral foramina as described in detail earlier[3].Inourpatients,thepermanentleadwasimplanted into the foramen with the best motor and sensory response inthetestingphase(T able1).Inthesecondstage,permanent pulse generator was implanted for the patients with signif- icant objective relief of symptoms after 3-4 weeks testing period with external pulse generator. In those with negative symptomrelief,theleadwasremovedunderlocalanesthesia. Aself-designeddisease-relatedquestionnairewasusedto assess pelvic pain symptoms as well as bowel and urogenital function before and 3–6 months after SNM procedure. CaseReportsinNeurologicalMedicine 3 Table2:Mainpre-SNMsymptomsandresponsetoSNMtreatment. Patient 1 2 3 TimepointtoSNM Pre 0.5yrs 2.5yrs Pre 0.5yrs 2.5yrs Pre 0.5yrs 2.5yrs Symptom Abdominalorpelvicpain yes 4 5 yes 4 4 yes 3 4 Dyspareunia yes 4 3 yes 4 3 yes na na Dyscheziaorbowelcolic yes 5 4 yes 4 4 no na na Constipation,outletobstruction yes 5 3 no na na yes 4 5 Analincontinence yes 5 4 yes 4 3 no na na Dysuria no na na yes 4 5 no na na Voidingdysfunction yes 4 2 no na na no na na Urinaryincontinence yes 4 2 yes 4 4 no na na SatisfactiontoSNM(NRS) 8 8 9 9 9 10 Comparedtopre-SNMstatus: 1:worse,2:nochange,3:somewhatimproved,4:muchimproved,5:excellentimprovement. NRS:numericalratingscale0–10. na:notapplicable. This questionnaire evaluated the presence of abdominal or pelvic pain or dyspareunia; functional bowel symptoms like pain and difficulties on defecation, fecal incontinence, or constipation; urinary symptoms like incomplete bladder emptying, dysuria, and urinary incontinence. In addition, patients were advised to keep daily pain diary and pain medication diary for fourteen days before, during, and after SNMtestperiod.Furthermore,womenwithpermanentpulse generatorwereaskedtoevaluatetheeffectofSNMtreatment topelvicpainsymptomsandbowelandbladderfunction3–6 months after procedure (1: worse, 2: no change, 3: somewhat improved, 4: much improved, 5: excellent improvement). Women’s subjective satisfaction on SNM was evaluated with n u m e r i c a lr a t i n gs c a l e( N R S )( 0m e a n i n gt o t a l l yu n s a t i s fi e d and 10 meaning totally satisfied). During the treatment and follow-up, women had the possibility for daily contact with studynurse. Main pre-SNM symptoms and self-reported response to treatmentareshowninTable2. Afterthetestperiod,threepatientsreportedconsiderable or excellent improvement (NRS 8-9) in subjective quality o fl i f ea n dw e r eo ff e r e dt oh a v ep e r m a n e n tp u l s eg e n e r a t o r installed. Two women found considerable improvement in bladder symptoms and two women found considerable or excellent relief in dyspareunia. All these three women found that SNM relieved chronic pelvic pain a little, but the pain waseasiertotoleratewhenfunctionalpelvicsymptomseased. Totaluseofpainmedicationdecreased,butonlyonewoman filledthequestionnairefullyadequatelyregardingNRSvalues andpainmedication. Patient number four did not experience any improve- ment in symptoms during the test period. Permanent pulse g e n e r a t o rw a sn o ti n s t a l l e d .S h ed i dn o tr e t u r na n yo ft h e questionnaires. We organized a phone call control to all four patients 2.5 years after the test period. All three patients with SNM installedwantedtocontinuewithSNM.Noneofthepatients with SNM were totally symptom-free but they all reported better quality of life by better symptom control enabling working, sociallife, andtravelling.Main pre-SNM symptom for patient 1 was disabling chronic pain and defecation problems. She got excellent help for pain and dyspareunia fromSNMbutthefirstmodulatorhadtoberemovedbecause ofinfection.Secondmodulatorgavegoodpaincontrolbutit has not been as good with dyspareunia. For good symptom control, she has needed SNM reprogramming 2-3 times a year. Patient 2 announced that chronic pelvic pain and pain during defecation had eased as well as fecal and urinary incontinence. Patient 3 had excellent help for obstipation and lived a regular life. She was fully satisfied with SNM. Fourth patient has retired in the age of 38 and suffers chronic pelvic pain, functional bowel symptoms, dysuria, and dyspareunia. No spontaneous symptom mitigation has occurred during 2.5 years. She underwent full clinical and radiological reevaluation with MRI year 2015 and has tried allhormonaltreatmentsandpainkillerswithouthelp. 3. Discussion Ourclinicalexperienceisthatdeependometrioticnodulesat area of uterosacral ligaments with infiltration to hypogastric nervesandtheureterscreatethemostdifficultdiseasetotreat. Possibilitiesofsurgeryarelimitedbypreservationofvascular, urinary, and neural structures. Nature of the pain is often neurogenicandresponsetohormonaltreatmentandregular pain medication is poor. Luckily, this end stage is rare but thosefewpatientsmayendupbeingquitedesperate. S N Mh a sb e e nu s e df o rp e l v i cfl o o rp a i na n df u n c t i o n a l pelvicdisordersforotherindicationsbutnotforendometrio- sis[9].WedecidedtoproposeSNMonexperimentalbasisfor wo menwhohadtriedallotherpossibletr ea tmen ts. A self-administered disease-related questionnaire was usedtoassessproblemswithbowelandurogenitalfunctions, but the compliance was poor. After the SNM test period, threeoutoffourpatientsannouncedsignificantreliefintheir symptoms and a permanent pulse generator was implanted. 4 CaseReportsinNeurologicalMedicine NRS did not turn out to be reliable indicator of pain since t h eh e a v yu s eo fp a i nm e d i c a t i o ni nt h eb e g i n n i n go fs t u d y b i a s e dt h er e s u l t s .H o w e v e r ,t h r e eo u to ff o u rp a t i e n t sw e r e able to reduce pain medication. These three women also felt that problems with defecation and urinary symptoms were alleviated significantly. In addition, two women got a remarkable help for dyspareunia and reported great increase insubjectivequalityoflife.Allthreepatientswanttocontinue with the SNM after 2.5 years. Fourth patient who did not benefitfromSNMhasnotexperiencedanyspontaneousrelief insymptomsduring2.5years. WearefullyawareoftheplaceboeffectofSNM.However, since three out of four patients with chronic pelvic pain and pelvic floor dysfunction reported a clinically plausible benefit of this pilot trial, we believe that SNM for end stage endometriosispatientsisworthfurthercontrolledstudies. Abbreviations SNM: Sacralneuromodulation NRS: Numericalratingscale DIE: Deepinfiltratingendometriosis NSAID: Nonsteroidalanti-inflammatorydrug. Additional Points Pr´ecis. Sacral neuromodulation may relieve pelvic pain and functional pelvic disorders in endometriosis patients resis- tanttosurgicalandmedicaltreatment. Competing Interests Theauthorsdeclarethattheyhavenocompetinginterests. Acknowledgments The authors are thankful to study nurse Marja-Liisa Mulo- Rantanen. References [ 1 ]A .M .J a d a v ,H .W a d h a w a n ,G .L .J o n e s ,L .W .W h e l d o n ,S . C. Radley, and S. R. Brown, “Does sacral nerve stimulation improve global pelvic function in women?”Colorectal Disease, vol.15,no .7 ,p p .848–857 ,2013. [2] J. Martellucci, G. Naldini, and A. Carriero, “Sacral nerve mod- ulation in the treatment of chronic pelvic pain,”International Journal of Colorectal Disease,vol.27 ,no .7 ,p p .9 21 –9 26,2012. [3] M. Spinelli and K.-D. Sievert, “Latest technologic and surgical developments in using InterStim/_0054therapy for sacral neuro- modulation:impactontreatmentsuccessandsafety,” European Urology,vol.54,no .6,p p .1287 –1296,2008. [ 4 ]K .N y a n g o hT i m o h ,G .C a n l o r b e ,D .V e r o l l e te ta l . ,“ C o n t r i b u - tion of sacral neuromodulation to manage persistent voiding dysfunction after surgery for deep infiltrating endometriosis with colorectal involvement: preliminary results,”European Journal of Obstetrics Gynecology and Reproductive Biology,v o l . 190,pp.31–35,2015. [5] L. C. Giudice, “Endometriosis,”New England Journal of Medicine,vol.36 2,no .25,p p .238 9–2398,2010. [6] S.-Z.Jia,J.-H.Leng,J.-H.Shi,P .-R.Sun,andJ.-H.Lang,“Health- relatedqualityoflifeinwomenwithendometriosis:asystematic review,”Journal of Ovarian Research,vol.5,no.1,article29,2012. [ 7 ]G .A .J .D u n s e l m a n ,N .V e r m e u l e n ,C .B e c k e re ta l . ,“ E S H R E guideline:managementofwomenwithendometriosis,” Human Reproduction,vol.29 ,no .3,p p .400–412,201 4. [8] I. S. Fraser, “Mysteries of endometriosis pain: Chien-Tien Hsu Memorial Lecture2009,”Journal of Obstetrics and Gynaecology Research,vol.36,no .1,p p .1 –10,2010. [ 9 ]R .K .L e o n g ,T .A .M a r c e l i s s e n ,F .H .N i e m a n ,R .A .D eB i e ,P . E. Van Kerrebroeck, and S. G. De Wachter, “Satisfaction and patient experience with sacral neuromodulation: results of a single center sample survey,”Journal of Urology,v o l .1 8 5 ,n o .2 , pp.588–592,2011. 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