Gruber, Daniel

Hope, Erica R.

English

George Washington University: Health Sciences Research Commons (HSRC)

Himmelfarb Health Sciences Library, The George Washington UniversityHealth Sciences Research CommonsObstetrics and Gynecology Faculty Publications Obstetrics and Gynecology12-2012Coccygeal fracture pain cured by sacralneuromodulation: A case reportErica R. HopeDaniel GruberGeorge Washington UniversityFollow this and additional works at: http://hsrc.himmelfarb.gwu.edu/smhs_obgyn_facpubsPart of the Obstetrics and Gynecology CommonsThis Letter to the Editor is brought to you for free and open access by the Obstetrics and Gynecology at Health Sciences Research Commons. It hasbeen accepted for inclusion in Obstetrics and Gynecology Faculty Publications by an authorized administrator of Health Sciences Research Commons.For more information, please contact hsrc@gwu.edu.Recommended CitationHope, E.R., Gruber, D.D. (2012). Coccygeal fracture pain caused by sacral neuromodulation: A case report. Neuromodulation:Technology at the Neural Interface, 16(6), 614-617.LETTER TO THE EDITORCoccygeal Fracture Pain Cured by SacralNeuromodulation: A Case ReportTo the Editor:There are many painful syndromes that are often difficult to treat.One option is electrotherapy, also known as neurostimulation. TheGate Control Theory first described by Melzack and Wall (1) in 1965postulated that nerves carrying painful peripheral stimuli andnerves carrying touch and vibratory sensation both terminate in thedorsal horn, which acts as the gate of the spinal cord. They hypoth-esized that input to these nerves could“close the gate”to the painfulstimuli, thereby eliminating pain.Based on the Gate Control Theory, Shealy et al. (2) in 1967 showedsuccess in treatment of chronic pain using the first spinal cordstimulator. Continuing this work, Shimoji et al. (3) in 1993 createdepidural spinal cord stimulation (SCS) that displayed analgesicproperties.SCS use has been widespread, and is Food and Drug Administra-tion (FDA) approved for numerous conditions. In addition tocervical, thoracic, and lumber stimulators, there is also sacralneuromodulation.Sacral neuromodulation (InterStim®, Medtronic, Minneapolis,MN) has been indicated by the FDA for the treatment of urinaryurgency, urinary frequency, and fecal incontinence. It is the onlyapproved sacral neuromodulator. However, very little data exist onits use for chronic pain or lower back pain. We describe a case whereInterStim was used to treat urinary urgency/frequency symptomsand incidentally relieved chronic lower back pain from a previouspelvic fracture.CASE REPORTA 74-year-old G2P2 postmenopausal female presented withlong-standing urge-dominant mixed urinary incontinence whopreviously failed two anticholinergic medications, tolterodine anddarifenacin. She also underwent one year of pelvic floor physicaltherapy with little improvement. She received an InterStim Periph-eral Nerve Stimulation Phase I with incision and implantation ofbilateral tine quadripolar lead electrodes into the S3 foraminaewith fluoroscopic guidance under conscious sedation with localanesthesia in December 2008. The placement was successful bypatient identification of sensation, great toe flexion, and bellowingof the anus noted. She experienced a suboptimal urgencyresponse (defined as <50% reduction in symptoms), but pro-ceeded to InterStim Phase II with incision and subcutaneousimplantation of sacral nerve neurostimulator with electronic analy-sis and complex programming with the left lead as that side had agreat reduction in symptoms. She elected to continue with theInterStim implantation because it significantly improved herchronic coccygeal pain that resulted from a motor vehicle-inducedpelvic fracture, 25 years prior. She was so used to this debilitatingpain, which sometimes required her to have to “crawl to the bath-room at night,” that she failed to inform us of this medical condi-tion prior to implantation. Her daily pain rating went from an8–10/10 to 2–4/10 on the visual analog scale. A sacral x-ray con-firmed correct placement of the lead at the S3 level on thatpatient’s left (Figs 1 and 2).She continued to have daily episodes of leakage of urine andbegan tolterodine again to help with urge symptoms, but greatlydesired the InterStim kept on because of considerable coccygealpain relief. The medication was subsequently discontinued as it didnot reduce her urge symptoms.The patient subsequently lost the remote device for the InterStimtwo months after placement and it was turned off for severalmonths. She noted prompt return of her chronic coccygeal painThe authors have no disclosures or conflicts of interest.The authors listed each had a substantial contribution to this case report.The views expressed in this article are those of the authors and do not reflect theofficial policy of the Department of the Army, Department of Defense, or the U.S.Government.Figure 1. Lateral sacral x-ray showing correct InterStim lead placement at S3level.Neuromodulation: Technology at the Neural InterfaceReceived: March 8, 2012 Revised: July 8, 2012 Accepted: September 3, 2012(onlinelibrary.wiley.com) DOI: 10.1111/ner.12002614www.neuromodulationjournal.com Neuromodulation 2013; 16: 614–617Published 2012. This article is a U.S. Government work and is in the public domain in the USA.after losing the remote. After presenting to the office and obtaininga new remote with the device turned on, the patient stated that sheagain felt immediate relief from her pain.She underwent collagen injections in November 2009 andJanuary 2010 (one year postimplant) for stress urinary inconti-nence symptoms, with significant improvement in stress and urgeurinary incontinence symptoms for one week and then worseningurge symptoms daily thereafter. The patient restarted pelvic floorphysical therapy and at the time stated that InterStim helps allevi-ate urge symptoms at night, but does not reduce daytime symp-toms. However, she still wants the InterStim kept on due to thesubstantial relief of her chronic coccygeal pain from the previouspelvic fracture.She subsequently had Botulinum-A Toxin (Botulinum-A Toxin(Botox®, Allergen Pharmaceuticals, Irvine, CA, USA) bladder injec-tions for worsening urge symptoms in March 2011 (2.5 yearspostimplant). As of May 2012 (3.5 years postimplant), she reportsthat her urge symptoms are significantly improved and hercoccygeal pain is still controlled with the continued use of theInsterStim.DISCUSSIONSCS has incomplete coverage for many areas of the body, includ-ing, but not limited to, low back, buttocks, feet, groin, pelvis, andneck. The S2-5 dermatomes are supplied by pathways that aresomatotopically deep in the spinal cord and are often times difficultto reach with SCS (4).Peripheral nerve stimulation works by activating inhibitory inter-neurons as well as primary afferent delta fibers, and has the poten-tial to recruit more nerve fibers than SCS. SCS has to penetratethrough multiple dural layers as well as cerebrospinal fluid, and theprimary afferent delta fibers are not accessible (5).Chronic pain disorders are more common among women thanmen. Women also experience increased frequency of pain as well ashigher disability rates and greater allocation of healthcare resources(5). Chronic pain in the sacral area has traditionally been difficult tocontrol, and the unique anatomy in this region may contribute.Many chronic pelvic pain conditions are suspected to be neuro-pathic in nature, including coccygodynia; however, this has notbeen proven (5). This is further supported by the fact that there areextremely limited medical and/or surgical interventions that allevi-ate pain in a majority of patients (6). Patients with pelvic pain typi-cally respond best to bilateral S2 or S3 electrodes as opposed tounilateral (4).Alo et al. in 1999 (7) (Table 1) used selective nerve root cathetersin the epidural space for five chronic pain patients who failedseveral types of conservative treatment and had at least oneabnormal objective finding. The chronic pain syndromes involvedilioinguinal neuralgia, discogenic pain at L4-5, failed back syn-drome, vulvodynia, and interstitial cystitis. Multielectrode systemswere placed in the thoracic, lumbar, and/or sacral areas. Allpatients had >50% reduction in pain level, with their visualanalogs declining significantly after the seven-day trial and allrequested permanent implantation of a nerve root stimulationsystem. They also postulated that sacral electrodes appeared lessvulnerable anatomically to patient movement than electrodes atother levels, possibly because they are contained within the rootsleeve in the spinal canal.Again in 1999, Alo et al. (8) reported a cohort of 80 patients with36-month follow-up after lead placement, primarily in thoracic andcervical regions with a majority diagnosed with low back andextremity pain. They were initially given a single program aftermultiple electrodes were placed, then given access for multipleprogram stimulation with patient control. This allowed patients tocontrol their level of success and alleviate the all-or-nothing phe-nomenon. Eighty percent of patients would have the procedureagain and 72% were still using the SCS system daily after 36months.In 2001, Alo and Mckay (9) published a case report of a 32-year-old woman with interstitial cystitis with intractable 10/10 pain thatseverely disrupted her quality of life. Two sacral nerve root stimula-tor electrodes were placed at the S2-3 level bilaterally. During herseven-day trial phase, she had increased bladder volumes,decreased pain, and stopped all bladder pain and spasm medica-tions. Her pain level was significantly reduced at 1/10 by the visualanalog scale. She subsequently underwent permanent implanta-tion at the same level. At publication, patient was one year afterimplantation, only requiring occasional use of the stimulator andhas rare symptoms, with her preinterstitial cystitis quality of liferestored. A literature search for coccygeal fracture or neuromodula-tion revealed one case report of a female that had previously under-gone InterStim implantation for urinary incontinence and also hadbilateral lower extremity pain and back pain from postlaminectomysyndrome (10). In this case, however, the InterStim did not improveher pain, but instead they used SCS at levels T8-T9-T10 which helpedboth her urinary incontinence and relieved her pain completely. Inaddition, they tried the InterStim vs. the SCS (turning one off andthen the other for two-month period) and they were equal in termsof urinary continence symptom relief, but only the SCS improvedboth her lower extremity and back pain.Figure 2. Anterior-posterior sacral x-ray confirming lead placement atpatient’s S3 level, left side.615LETTER TO THE EDITORwww.neuromodulationjournal.com Neuromodulation 2013; 16: 614–617Published 2012. This article is a U.S. Government work and is in the public domain in the USA.Several papers referred to postoperative pain in terms of painafter placement and did not refer to preoperative pain that wasrelieved with InterStim. Pauls et al. reported a prospective studythat evaluated sexual function after InterStim implantation forurinary incontinence; however, patients did not have a history ofchronic pelvic pain (11). There was a significant improvement in painduring intercourse (p = 0.015).Marinkovic et al. reported patients with interstitial cystitis andpainful bladder syndrome that had a significant improvement inpain after sacral neuromodulation (p < 0.01) (12). Another paperon interstitial cystitis by Peters et al. describes a moderate ormarked improvement in pain after InterStim implantation in treat-ing chronic pain associated with interstitial cystitis refractory tostandard therapy (13). Finally, another paper by Peters et al. foundthat 69% of their patients reported moderate or marked improve-ment in pelvic pain also with interstitial cystitis (14).Only one case report was found that discussed SCS for treatmentof coccygodynia after a coccygeal fracture, by Yakovlev and Resch(15). A 37-year-old male with a two-year history of coccygodyniaafter a closed sacral and coccygeal fracture as well as a rectal tear. Hereceived surgery for the perineal wound, but continued to experi-ence pain after the fractures had healed. Two epidural leads wereplaced with a satisfactory trial response. The final leads were placedat T8, T9, and T10, with excellent pain relief reported. The pain nolonger required pain medications and his quality of life improved.Our patient underwent a lateral sacral x-ray which confirmedcorrect lead placement at the S3 foramina; however, the pudendaland coccygeal nerve roots are closely related. The S3 nerve rootcontributes to the pudendal nerve. S4 and S5 nerve roots supply thecoccyx, anus, and rectal areas. It is possible that our patient hasvariant anatomy and that the S3 nerve root supplies a portion of thecoccygeal area, which would account for her pain relief associatedwith the InterStim device.The majority of pain that the sacral neuromodulation has previ-ously treated has been chronic pelvic pain that is refractory to othertherapies, which often coexists with urinary incontinence or refrac-tory interstitial cystitis. For these two indications, it appears that thesacral neuromodulation has a significant improvement in pain. Nocitations were found that described the use of sacral neuromodula-tion in terms of coccygeal pain; only SCS has previously been used.In conclusion, sacral neuromodulation has the potential for treat-ment of coccygeal pain.Authorship StatementDr. Gruber had the idea for the case report and took care of thepatient in the clinic setting. Dr. Hope prepared the case report draftwith important intellectual input from Dr. Gruber. All authorsapproved the final case report/letter.Erica R. Hope, MD, Daniel D. Gruber, MDDepartment of Obstetrics & Gynecology, Division of Female PelvicMedicine & Reconstructive Surgery, Walter Reed National Military MedicalCenter, Bethesda, MD, USAREFERENCES1. Melzack R, Wall PD. Pain mechanisms: a new theory. Science 1965;150:971–979.2. Shealy CN, Mortimer JT, Reswick JB.Electrical inhibition of pain by stimulation of thedorsal columns: preliminary clinical report. Anesth Analg 1967;46:489–491.3. Shimoji K,Hokari T,Kano T et al.Management of intractable pain with percutaneousepidural spinal cord stimulation: differences in pain-relieving effects among dis-eases and sites of pain. Anesth Analg 1993;77:110–116.4. Stuart RM, Winfree CJ. Neurostimulation techniques for painful peripheral nervedisorders. Neurosurg Clin N Am 2009;20:111–120.5. Kothari S. Neuromodulatory approaches to chronic pelvic pain and coccygodynia.Acta Neurochir Suppl 2007;97:365–371.6. Alo K, Holsheimer J. New trends in neuromodulation for the management of neu-ropathic pain. Neurosurgery 2002;50:690–703.7. Alo KM, Yland MJ, Redko V et al. Lumbar and sacral nerve root stimulation (NRS) inthe treatment of chronic pain: a novel anatomic approach and neurostimulationtechnique. Neuromodulation 1999;2:23–31.8. Alo KM, Yland MJ, Charnov JH, Redko V. Multiple program spinal cord stimulation inthe treatment of chronic pain: follow-up of multiple program SCS. Neuromodulation1999;2:266–272.9. Alo KM, Mckay E. Selective nerve root stimulation (SNRS) for the treatment of intrac-table pelvic pain and motor dysfunction: a case report. Neuromodulation 2001;4:19–23.10. Yakovlev AE, Resch BE. Treatment of urinary voiding dysfunction syndromes withspinal cord stimulation. Clin Med Res 2010;8:22–24.Table 1. Literature Search Description and Outcomes.Author Year Description OutcomeAlo et al. (7) 1999 Selective nerve root catheters for chronic pain in thoracic,lumbar, and sacral areas>50% reduction in VAS pain level; all had permanentimplantationAlo et al. (8) 1999 80 patients with thoracic and cervical leads for low backand extremity pain; single vs. multiple program controls36-month follow-up, with 72% of patients still using systemdailyAlo and Mckay (9) 2001 Intractable interstitial cystitis pain with sacralneuromodulator placedSignificantly improved quality of life; rare symptoms withoccasional use of stimulatorMarinkovic et al.(12)2011 Retrospective, case control to evaluate sacralneuromodulation in interstitial cystitis patientsStatistically significant (p < 0.01) improvement in urinaryfrequency and visual analog pain scale scores at 6+ yearfollow-upPauls et al. (11) 2004 Refractory interstitial cystitis with chronic pelvic painpatients treated with sacral neuromodulationDecreased narcotic requirements and subjective pelvic painPeters et al. (14) 2003 Refractory interstitial cystitis treated with sacralneuromodulation via percutaneous approach or stagedprocedureSymptom and quality of life improvement; improvedimplantation rate with staged procedure (94% vs. 52%)Peters et al. (13) 2004 Refractory interstitial cystitis treated with sacralneuromodulation69% patients with moderate or marked improvement inpelvic painYakovlev andResch (10)2010 InterStim implantation and SCS for urinary incontinenceand postlaminectomy syndromeInterStim and SCS equal in incontinence relief; SCSimproved lower extremity and back pain alsoYakovlev andResch (15)2012 Spinal cord stimulation after coccygeal fracture withcoccygodyniaT8-T10 leads with excellent pain relief; no longer requiringdaily pain medicationSCS, spinal cord stimulation; VAS, visual analog scale.616www.neuromodulationjournal.com Neuromodulation 2013; 16: 614–617Published 2012. This article is a U.S. Government work and is in the public domain in the USA.11. Pauls RN, Marinkovic SP, Silva WA et al. Effects of sacral neuromodulation on femalesexual function. Int Urogynecol J 2007;18:391–395.12. Marinkovic SP, Gillen LM, Marinkovic CM. Minimum 6-year outcomes for interstitialcystitis treated with sacral neuromodulation. Int Urogynecol J 2011;22:407–412.13. Peters KM, Carey J, Konstandt D. Sacral neuromodulation decreases narcoticrequirements in refractory interstitial cystitis: outcomes based on technique. BJU Int2004;93:777–779.14. Peters KM, Carey JM, Konstandt D. Sacral neuromodulation decreases narcoticrequirements in refractory interstitial cystitis. Int Urogynecol J 2003;14:223–228.15. Yakovlev A, Resch BE. Spinal cord stimulation for the treatment of coccydynia fol-lowing traumatic closed fracture of sacrum and coccyx and rectal tear. Pain Pract2012;1 (Suppl. 12):182.Keywords: coccygeal fracture, coccygeal pain, InterStim,neuromodulation617LETTER TO THE EDITORwww.neuromodulationjournal.com Neuromodulation 2013; 16: 614–617Published 2012. This article is a U.S. Government work and is in the public domain in the USA.

Coccygeal fracture pain cured by sacral neuromodulation: A case report

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Coccygeal fracture pain cured by sacral neuromodulation: A case report

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