{"paper_id":"4eafd217-ac9c-4fd6-9c74-38a95e5d1dab","body_text":"Case Report\nSacral Neuromodulation: Foray into Chronic Pelvic Pain in\nEnd Stage Endometriosis\nMaija Lavonius, Pia Suvitie, Pirita Varpe, and Heikki Huhtinen\nDepartment of Digestive Surgery and Department of Gynecology and Obstetrics, Turku University Hospital, Turku, Finland\nCorrespondenceshouldbeaddressedtoMaijaLavonius;maija.lavonius@tyks.fi\nReceived 28 August 2016; Revised 11 January 2017; Accepted 19 February 2017; Published 6 March 2017\nA\ncademicEditor:DominicB.Fee\nCopyright © 2017 MaijaLavoniusetal.ThisisanopenaccessarticledistributedundertheCreativeCommonsAttributionLicense,\nwhichpermitsunrestricteduse,distribution,andreproductio ninanymedium,providedtheoriginalworkisproperlycited.\nExcisionofallendometrioticlesionsisthemethodofchoiceinthetreatmentofsevereendometriosisresistanttomedicaltherapy.\nTheinfiltratingnatureofthediseaseaswellasextensivesurgerymay,however,causechronicpainthatcannotberelievedbyeither\nsurgeryorhormonaltreatment.Asapilottreatment,wetestedtheeffectofsacralneuromodulation(SNM)forfourendometriosis\npatients suffering chronic pelvic pain and pelvic organ dysfunction after radical surgical treatment. Three out of four patients\nreported improvement in their symptoms during the neuromodulation testing period and a permanent pulse generator was\ninstalled. After 2.5 years, all three patients report better quality of life and want to continue with SNM.\n1. Introduction\nSacral neuromodulation (SNM) involves electrical modula-\ntion of a sacral nerve root by means of an electrode and a\npulse generator. The conventional indications for SNM are\nurinary retention, urinary incontinence, anal incontinence,\nand constipation [1]. Several studies have also demonstrated\npromisingresultsofSNMinchronicpelvicpain[2],although\nits mechanism of action remains ill defined [3]. One study\nhasevalua tedtheeffectofSNMonvoidingdysfunctionafter\nsurgeryfordeepinfiltrating(DIE)endometriosis[4].Toour\nknowledge,noreportsofSNMforthetreatmentofpelvicpain\nafterradicalsurgeryofendometriosishavebeenpublished.\nEndometriosisisanestrogen-dependentchronicinflam-\nmatory disease defined by the presence of functional endo-\nm 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 –\n10%ofwomenareaffectedduringtheirfertileyearssuffering\nfrom a variety of pelvic pain symptoms and subfertility [5].\nEndometriosis significantly reduces health-related quality of\nlife (HRQoL) mostly due to pain symptoms [6]. Pain can be\nalleviated with hormonal medication and nonsteroidal anti-\ninflammatory drugs (NSAID) [7]. When medical therapy\nfails, complete surgical excision of the endometriotic lesions\nis the method of choice. In severe DIE, a multidisciplinary\na p p r oachcanbenecessary .\nEndometriosis can cause pain in many different mech-\nanisms [8]. Irritation of pelvic nerves due to inflammation\ncausesnociceptivepain.Inaddition,DIEcaninfiltratepelvic\nnerves causing pain, and de novo nerve growth is also\ndetected in ectopic endometrial tissue. During surgery, all\nefforts are made to preserve the pelvic nerves. However,\nin severe cases, nerve-sparing technique is not possible if\nthe goal is to remove all endometriosis. In such cases, a\ncompromise is made between radicalness of surgery and\npreservation of pelvic organ function. The disease itself as\nwell as extensive surgery can lead to chronic neuropathic\npain.Insuchcases,littleoptionsareavailabletoeasethepain.\n2. Case Presentation\nCommon to all our four patients was a history of sur-\ngically treated deep infiltrating endometriosis in lateral\npelvicsidewallsaffectingureters,sacrouterineligaments,and\npelvic nerves. All women had undergone hysterectomy and\nbilateral salpingo-oophorectomy combined with excision of\nall macroscopic endometriosis. Thus, adenomyosis as the\nsource of pain was excluded. No recurrent endometriosis\nwassuspectedingynecologicalexaminationandtransvaginal\nultrasound, and the chronic noncyclical pelvic pain was life\ninterferingandresistanttohormonalandmedicaltreatment.\nHindawi\nCase Reports in Neurological Medicine\nVolume 2017, Article ID 2197831, 4 pages\nhttps://doi.org/10.1155/2017/2197831\n\n2 CaseReportsinNeurologicalMedicine\nTable1:Surgicalhistoryofthefourwomenintheneuromodulationpilotstudy.Operationswereperformedbylaparotomyiflaparoscopyis\nnotmentionedinthetext.\nPatient Age\n(years) year Operation Indication Postoperative\ncomplications\n1\nG2P2\n1999 Laparoscopy Endometriosis None\n2000 Laparoscopicleftsalpingo-oophorectomy Endometriosis None\n2000 Laparoscopic right salpingo-oophorectomy,\nsupracervicalhysterectomy Endometriosis None\n2009 Adhesiolysisandextirpationoftheuterinecervix Pelvicpain None\n2011 Anteriorresection,appendicectomy,and\nadhesiolysis Pelvicpain(leftside) None\n43 2014 Neuromodulationtestperiodandapermanentpulse\ngeneratorinstallation(rightS4) Pelvicpain None\n2\nG0P0\n2002 Leftsalpingo-oophorectomyandadhesiolysis Endometriosis None\n2006 Anteriorresectionandileostomy Sigmoidperforation Rectalanastomosis\nstricture\n2007 Reanteriorresectionandileostomyclosure None\n2009 Adhesiolysis,ileumresection,andsupracervical\nhysterectomy\nPostoperativeadhesions,\nbowelobstruction None\n2013 Re-reanteriorresection,resectionofleftureter,\nextirpationoftheuterinecervix,andileostomy\nEndometriosis(main\nsymptompain)andanal\nincontinenceand\ndefecationdifficulties\nUreteralanastomosis\nleakagetreatedby\npyelostomyanddrainage\n2013 Ileostomyclosure None\n50 2014 Neuromodulationtestperiodandapermanentpulse\ngeneratorinstallation(leftS4)\nPelvicpainanddefecation\ndifficulties None\n3\nG2P2\n1991 Laparoscopy Endometriosis None\n1995 Laparoscopy Endometriosis None\n2003 Laparoscopicexcisionofsacrouterineligamentsand\nelectrocoagulationofperitonealendometriosis Endometriosis None\n2005 Laparoscopicelectrocoagulationofperitoneal\nendometriosisandadhesiolysis Endometriosis None\n2008 Hysterectomyandexcisionofperitoneal\nendometriosis Endometriosis None\n2010 Laparoscopicadhesiolysisandleft\nsalpingo-oophorectomy\nPostoperativeadhesions,\npain,leftovariancyst None\n2010 Vaginal resection, extirpation of left periureteral\nendometriosis\nEndometriosis,\nhydronephrosis\nLaparotomywound\ninfection\n42 2014 Neuromodulationtestingperiodandapermanent\npulsegeneratorinstallation(rightS4)\nPelvicpainanddefecation\ndifficulties None\n4\nG1P0\n2011 Hysterectomy andbilateral salpingectomy Endometriosis None\n2012\nIleumresection,appendicectomy,bilateral\noophorectomy,vaginalresection,andresectionof\nleftsacrouterineligament\nEndometriosis,pelvicpain None\n35 2014 Neuromodulationtestingperiod(leftS3) Pelvicpain None\nNote.G:gra vidi ty ,P:pari ty .\nDetailed surgical history is presented in Table 1. All women\nwere fully informed of the experimental nature of the SNM\ntreatmentforthisindication.Noethicsapprovalwasapplied\nbecause of acknowledged use of SNM for other indications\nwith pelvic disorders. SNM was offered to one more patient\nbutshedeclinedbecauseshefearedtheprocedure.\nThe SNM procedure with InterStim II/_0054system was\nperformed in two stages. In the first stage, a permanent\nlead was implanted under local anesthesia with fluoroscopic\ncontrol into S3 or S4 sacral foramina as described in detail\nearlier[3].Inourpatients,thepermanentleadwasimplanted\ninto the foramen with the best motor and sensory response\ninthetestingphase(T able1).Inthesecondstage,permanent\npulse generator was implanted for the patients with signif-\nicant objective relief of symptoms after 3-4 weeks testing\nperiod with external pulse generator. In those with negative\nsymptomrelief,theleadwasremovedunderlocalanesthesia.\nAself-designeddisease-relatedquestionnairewasusedto\nassess pelvic pain symptoms as well as bowel and urogenital\nfunction before and 3–6 months after SNM procedure.\n\nCaseReportsinNeurologicalMedicine 3\nTable2:Mainpre-SNMsymptomsandresponsetoSNMtreatment.\nPatient 1 2 3\nTimepointtoSNM Pre 0.5yrs 2.5yrs Pre 0.5yrs 2.5yrs Pre 0.5yrs 2.5yrs\nSymptom\nAbdominalorpelvicpain yes 4 5 yes 4 4 yes 3 4\nDyspareunia yes 4 3 yes 4 3 yes na na\nDyscheziaorbowelcolic yes 5 4 yes 4 4 no na na\nConstipation,outletobstruction yes 5 3 no na na yes 4 5\nAnalincontinence yes 5 4 yes 4 3 no na na\nDysuria no na na yes 4 5 no na na\nVoidingdysfunction yes 4 2 no na na no na na\nUrinaryincontinence yes 4 2 yes 4 4 no na na\nSatisfactiontoSNM(NRS) 8 8 9 9 9 10\nComparedtopre-SNMstatus:\n1:worse,2:nochange,3:somewhatimproved,4:muchimproved,5:excellentimprovement.\nNRS:numericalratingscale0–10.\nna:notapplicable.\nThis questionnaire evaluated the presence of abdominal\nor pelvic pain or dyspareunia; functional bowel symptoms\nlike pain and difficulties on defecation, fecal incontinence,\nor constipation; urinary symptoms like incomplete bladder\nemptying, dysuria, and urinary incontinence. In addition,\npatients were advised to keep daily pain diary and pain\nmedication diary for fourteen days before, during, and after\nSNMtestperiod.Furthermore,womenwithpermanentpulse\ngeneratorwereaskedtoevaluatetheeffectofSNMtreatment\ntopelvicpainsymptomsandbowelandbladderfunction3–6\nmonths after procedure (1: worse, 2: no change, 3: somewhat\nimproved, 4: much improved, 5: excellent improvement).\nWomen’s subjective satisfaction on SNM was evaluated with\nn 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\nand 10 meaning totally satisfied). During the treatment and\nfollow-up, women had the possibility for daily contact with\nstudynurse.\nMain pre-SNM symptoms and self-reported response to\ntreatmentareshowninTable2.\nAfterthetestperiod,threepatientsreportedconsiderable\nor excellent improvement (NRS 8-9) in subjective quality\no 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\ninstalled. Two women found considerable improvement in\nbladder symptoms and two women found considerable or\nexcellent relief in dyspareunia. All these three women found\nthat SNM relieved chronic pelvic pain a little, but the pain\nwaseasiertotoleratewhenfunctionalpelvicsymptomseased.\nTotaluseofpainmedicationdecreased,butonlyonewoman\nfilledthequestionnairefullyadequatelyregardingNRSvalues\nandpainmedication.\nPatient number four did not experience any improve-\nment in symptoms during the test period. Permanent pulse\ng 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\nquestionnaires.\nWe organized a phone call control to all four patients\n2.5 years after the test period. All three patients with SNM\ninstalledwantedtocontinuewithSNM.Noneofthepatients\nwith SNM were totally symptom-free but they all reported\nbetter quality of life by better symptom control enabling\nworking, sociallife, andtravelling.Main pre-SNM symptom\nfor patient 1 was disabling chronic pain and defecation\nproblems. She got excellent help for pain and dyspareunia\nfromSNMbutthefirstmodulatorhadtoberemovedbecause\nofinfection.Secondmodulatorgavegoodpaincontrolbutit\nhas not been as good with dyspareunia. For good symptom\ncontrol, she has needed SNM reprogramming 2-3 times a\nyear. Patient 2 announced that chronic pelvic pain and pain\nduring defecation had eased as well as fecal and urinary\nincontinence. Patient 3 had excellent help for obstipation\nand lived a regular life. She was fully satisfied with SNM.\nFourth patient has retired in the age of 38 and suffers\nchronic pelvic pain, functional bowel symptoms, dysuria,\nand dyspareunia. No spontaneous symptom mitigation has\noccurred during 2.5 years. She underwent full clinical and\nradiological reevaluation with MRI year 2015 and has tried\nallhormonaltreatmentsandpainkillerswithouthelp.\n3. Discussion\nOurclinicalexperienceisthatdeependometrioticnodulesat\narea of uterosacral ligaments with infiltration to hypogastric\nnervesandtheureterscreatethemostdifficultdiseasetotreat.\nPossibilitiesofsurgeryarelimitedbypreservationofvascular,\nurinary, and neural structures. Nature of the pain is often\nneurogenicandresponsetohormonaltreatmentandregular\npain medication is poor. Luckily, this end stage is rare but\nthosefewpatientsmayendupbeingquitedesperate.\nS 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\npelvicdisordersforotherindicationsbutnotforendometrio-\nsis[9].WedecidedtoproposeSNMonexperimentalbasisfor\nwo menwhohadtriedallotherpossibletr ea tmen ts.\nA self-administered disease-related questionnaire was\nusedtoassessproblemswithbowelandurogenitalfunctions,\nbut the compliance was poor. After the SNM test period,\nthreeoutoffourpatientsannouncedsignificantreliefintheir\nsymptoms and a permanent pulse generator was implanted.\n\n4 CaseReportsinNeurologicalMedicine\nNRS did not turn out to be reliable indicator of pain since\nt 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\nb 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\nable to reduce pain medication. These three women also\nfelt that problems with defecation and urinary symptoms\nwere alleviated significantly. In addition, two women got a\nremarkable help for dyspareunia and reported great increase\ninsubjectivequalityoflife.Allthreepatientswanttocontinue\nwith the SNM after 2.5 years. Fourth patient who did not\nbenefitfromSNMhasnotexperiencedanyspontaneousrelief\ninsymptomsduring2.5years.\nWearefullyawareoftheplaceboeffectofSNM.However,\nsince three out of four patients with chronic pelvic pain\nand pelvic floor dysfunction reported a clinically plausible\nbenefit of this pilot trial, we believe that SNM for end stage\nendometriosispatientsisworthfurthercontrolledstudies.\nAbbreviations\nSNM: Sacralneuromodulation\nNRS: Numericalratingscale\nDIE: Deepinfiltratingendometriosis\nNSAID: Nonsteroidalanti-inflammatorydrug.\nAdditional Points\nPr´ecis. Sacral neuromodulation may relieve pelvic pain and\nfunctional pelvic disorders in endometriosis patients resis-\ntanttosurgicalandmedicaltreatment.\nCompeting Interests\nTheauthorsdeclarethattheyhavenocompetinginterests.\nAcknowledgments\nThe authors are thankful to study nurse Marja-Liisa Mulo-\nRantanen.\nReferences\n[ 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 .\nC. Radley, and S. R. Brown, “Does sacral nerve stimulation\nimprove global pelvic function in women?”Colorectal Disease,\nvol.15,no .7 ,p p .848–857 ,2013.\n[2] J. Martellucci, G. Naldini, and A. Carriero, “Sacral nerve mod-\nulation in the treatment of chronic pelvic pain,”International\nJournal of Colorectal Disease,vol.27 ,no .7 ,p p .9 21 –9 26,2012.\n[3] M. Spinelli and K.-D. 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De Wachter, “Satisfaction and\npatient experience with sacral neuromodulation: results of a\nsingle center sample survey,”Journal of Urology,v o l .1 8 5 ,n o .2 ,\npp.588–592,2011.\n\nSubmit your manuscripts at\nhttps://www.hindawi.com\nStem Cells\nInternational\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nM ED IATO R S\nINFLAM MATION\nof\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nBehavioural \nNeurology\nEndocrinology\nInternational Journal of\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nDisease Markers\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nBioMed \nResearch International\nOncology\nJournal of\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nOxidative Medicine and \nCellular Longevity\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nPPAR Research\nThe Scientific \nWorld Journal\nHindawi Publishing Corporation \nhttp://www.hindawi.com Volume 2014\nImmunology Research\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nJournal of\nObesity\nJournal of\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\n Computational and  \nMathematical Methods \nin Medicine\nOphthalmology\nJournal of\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nDiabetes Research\nJournal of\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nResearch and Treatment\nAIDS\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nGastroenterology \nResearch and Practice\nHindawi Publishing Corporation\nhttp://www.hindawi.com Volume 2014\nParkinson’s \nDisease\nEvidence-Based \nComplementary and \nAlternative Medicine\nVolume 2014\nHindawi Publishing Corporation\nhttp://www.hindawi.com","source_license":"CC0","license_restricted":false}