Akashi Yoshimasa

Doi Manami

Enomoto Tsuyoshi

Furuya Kinji

Hamada Hiromi

Hashimoto Shinji

Hisakura Katsuji

Kim Jaejeong

Obata Mana

Oda Tatsuya

Ogawa Koichi

Ohara Rena

Ohara Yusuke

Owada Yohei

Shimomura Osamu

Takahashi Kazuhiro

Frontiers in Surgery

English

Tsukuba Repository  (Tulips-R)

CASE REPORTpublished: 24 June 2021doi: 10.3389/fsurg.2021.637719Frontiers in Surgery | www.frontiersin.org 1 June 2021 | Volume 8 | Article 637719Edited by:Vincenzo Neri,University of Foggia, ItalyReviewed by:Noushif M,Aster MIMS Hospital, IndiaAli Coskun,Izmir Bozyaka Training and ResearchHospital, Turkey*Correspondence:Yusuke Oharay.ohara@md.tsukuba.ac.jpSpecialty section:This article was submitted toVisceral Surgery,a section of the journalFrontiers in SurgeryReceived: 04 December 2020Accepted: 31 May 2021Published: 24 June 2021Citation:Ohara Y, Enomoto T, Owada Y,Hisakura K, Akashi Y, Ogawa K,Doi M, Takahashi K, Shimomura O,Furuya K, Kim J, Hashimoto S,Ohara R, Obata-Yasuoka M,Hamada H and Oda T (2021)Rectoperineal Fistula Presented 5Months After Repair of SevereObstetric Perineal Laceration: A CaseReport. Front. Surg. 8:637719.doi: 10.3389/fsurg.2021.637719Rectoperineal Fistula Presented 5Months After Repair of SevereObstetric Perineal Laceration: ACase ReportYusuke Ohara 1*, Tsuyoshi Enomoto 1, Yohei Owada 1, Katsuji Hisakura 1,Yoshimasa Akashi 1, Koichi Ogawa 1, Manami Doi 1, Kazuhiro Takahashi 1,Osamu Shimomura 1, Kinji Furuya 1, Jaejeong Kim 1, Shinji Hashimoto 1, Rena Ohara 2,Mana Obata-Yasuoka 2, Hiromi Hamada 2 and Tatsuya Oda 11Department of Gastrointestinal and Hepato-Biliary-Pancreatic Surgery, Faculty of Medicine, University of Tsukuba, Tsukuba,Japan, 2Department of Obstetrics and Gynecology, Faculty of Medicine, University of Tsukuba, Tsukuba, JapanIntroduction: Obstetric severe perineal laceration can frequently occur as a surgical siteinfection (SSI), which sometimes leads to rectovaginal fistula after repair. We encountereda rare case of a rectoperineal fistula 5 months after repair of a severe perineal laceration.Case presentation: The patient was a 39-year-old woman who underwent repair of afourth-degree perineal laceration after vaginal delivery. Five months after primary repair,she presented with perineal swelling and pain followed by uncontrollable flatulence orpassage of feces at the perineum, which was finally diagnosed as a rectoperineal fistula.Transperineal repair with fistulous tract excision was performed for the rectoperinealfistula. Closure of the rectum, perineal body, and vagina was performed layer-by-layerconstructing a thick perineum to prevent anal dysfunction. The fistula was successfullyclosed, and the patient did not show any symptoms of fecal incontinence 6 monthsafter surgery.Discussion: As the rectoperineal fistula might have resulted in SSI at the primaryrepair of the obstetric injury, the delayed occurrence of the rectoperineal fistula wasunusual. A perineal approach should be performed for complete fistulous tract excision,reconstruction of a robust perineal structure, and preservation of anal sphincter function.Keywords: rectoperineal fistula, obstetric laceration, anal incontinence, surgical site infection, gastrointestinalsurgeryINTRODUCTIONObstetric perineal lacerations, as 53-79% of women sustain them, are often accompanied by vaginaldelivery (1, 2). Traditionally, obstetric perineal laceration is classified into four degrees according tothe anatomical structure involved (3). Third-degree laceration extends to the anal external and/orinternal sphincter. Fourth-degree laceration extends to the rectal mucosa. These severe lacerationswith injury to the anal sphincter occurs in 6.4% of women (4), leading to fecal incontinence.Careful recognition and adequate repair of lacerations are essential for preventing postpartumanal incontinence.Ohara et al. Rectoperineal Fistula After Obstetric LacerationThe purpose of repairing a third- or fourth-degree lacerationis to restore the continuity of both the external and internal analsphincters (5). Moreover, a thick perineal body and rectovaginalseptum should be constructed to provide muscular and structuralsupport in the thin area between the anterior anorectal regionand vagina. Multilayer repair is optimal for severe laceration,maintaining the strength of perianal tissue, and preserving analcontinence (3).The repair of these severe lacerations carries a potential risk ofsurgical site infection (SSI), as the wound is easily contaminatedby intestinal or epidermal bacteria. Goldaber et al. reported that3.6% of patients with fourth-degree lacerations developed SSIafter the repair (6). Venkatesh et al. documented that 101 of2,500 women who underwent vaginal delivery presented withanorectal complications (7). They reported that 25 patients hada rectovaginal fistula; in contrast, only two patients (0.08%) hadan anoperineal fistula. Here, we report a rare case of delayedrectoperineal fistula 5 months after repair of a fourth-degreeperineal laceration resulting from a vaginal delivery.CASE PRESENTATIONA 39-year-old woman with a history of cesarean section for firstchildbirth was admitted to the University of Tsukuba Hospitalfor a second pregnancy. At 40 weeks of gestation for postdatepregnancy, a left lateral episiotomy followed by a forceps deliverywas performed, and a baby weighing 3.93 kg was born ingood condition. A fourth-degree perineal laceration was foundon perineal examination immediately after delivery. Briefly, avaginal examination showed a 7 cm laceration in the episiotomy.Rectal examination showed a 3 cm laceration in the lower rectum,FIGURE 1 | Pre-operative examination for the diagnosis of rectoperineal fistula (A) Magnetic resonance imaging (sagittal view) showing a small tract from the lowerrectum. Arrow, small tract. Arrowhead, rectum. (B) Small fistula on the perineal skin visualized after insertion of indigo carmine poured into the rectum (arrow).Arrowhead, vaginal orifice.but it did not advance to the anal ring. The external and internalsphincter injuries were minor.The first operation for perineal laceration was performedunder spinal anesthesia in the lithotomy position on the dayof delivery. A multilayer closure was performed followed byindependent sutures of the rectal wall, musculature of theperineal body with sphincter, vaginal epithelium, and skin. Allthe sutures were interrupted stitches using 4-0 monofilamentabsorbable sutures. Antibiotics (cefmetazole) were administeredprior to the surgery and used until 5 days after surgery. Wounddehiscence was not observed, and the patient was discharged 11days after surgery. The patient did not show any symptoms offecal incontinence, SSI, or rectovaginal fistula on the outpatientexamination 1 month after surgery.Five months after the first surgery, the patient noticed perinealswelling and pain, followed by an uncontrollable passage of gasor feces at the perineum. A small pinhole with a discharge wasnoted at the left perineum, at the 5-O’clock direction of thevagina, and 5mm from the vaginal orifice. Magnetic resonanceimaging revealed a small fistula tract from the lower rectum,but an external opening or perianal abscess was not revealed(Figure 1A). Colonoscopy also showed a small orifice at theanterior wall of the lower rectum (above the dentate line),and indigo carmine poured into the rectum was dischargedfrom the perineal opening (Figure 1B). Vaginoscopy did notreveal a fistula tract in the vagina. Finally, a rectoperineal fistulawas diagnosed. No evidence of Crohn’s disease was observed.Anatomical schema demonstrating the position of fistula tractwas shown in Figure 2.Seven months after the first operation, a second operation,transperineal repair for rectoperineal fistula was performedFrontiers in Surgery | www.frontiersin.org 2 June 2021 | Volume 8 | Article 637719Ohara et al. Rectoperineal Fistula After Obstetric LacerationFIGURE 2 | Anatomical schema of the rectoperineal fistula. (A) Sagittal image showing the position of fistula tract (arrow). (B) Image of perineal surface. The fistulaopened nearby the vaginal orifice. Perineal approach with transverse incision was placed at the surgery (dotted arrow).(Figures 2B, 3). Briefly, the patient received general anesthesiain the lithotomy position. A Lone Star Retractor System(CooperSurgical, Inc., Trumbull, USA) was attached to theanus and vagina. A 2mm probe was advanced through therectoperineal fistula to guide the route of the fistula. A 4 cmtransverse incision was made on the perineal skin, 5mmfrom the vaginal orifice. Dissection of the rectovaginal septumwas performed widely, separating the vagina and rectum, and2 cm of the entire fistulous tract was excised. A three-layerclosure (rectum, perineal body, and vagina, respectively) wasperformed with interrupted sutures using 4-0 monofilamentabsorbable sutures. The subcutaneous tissue and skin weresutured horizontally; thus, the incision was changed in thevertical direction, keeping thick perineal tissue. A trans-analdrain was placed until 5 days after surgery. Antibiotics (flomoxefsodium) were administered prior to the surgery and useduntil 3 days after surgery. The patient was discharged 10 daysafter surgery. The patient did not show any symptoms of SSI,rectovaginal fistula, or fecal incontinence on the outpatientexamination 6 months after surgery. Anorectal manometryshowed a normal anal sphincter function. Colonoscopy showedno dip or fistula on the rectal mucosa.DISCUSSIONPresent case exhibited two unusual features. First, the fistulatract due to severe perineal laceration advanced to perineal skin,not to anorectum. Second, the rectoperineal fistula presentedsymptoms several months after initial repairment. These factorswould remarkably affect surgical strategy and post-operativesurveillance of severe obstetric lacerations.Perineal wound complications after severe obstetric perineallacerations are uncommon, but they can lead to significantmorbidity including chronic pain, incontinence, loss of sexualfunction, and rectovaginal fistula (8, 9). Rectovaginal fistula aftervaginal delivery can result from a failure to recognize the primaryobstetric injury, inadequate repair, or surgical site infection of arepaired wound. Rectoperineal fistula, which might be similarto rectovaginal fistula, is a rare complication even after severeperineal lacerations. Lewicky-Gaupp et al. demonstrated the riskof wound complications in a prospective cohort that enrolled 268women with obstetric anal sphincter injury. They showed that53 (19.8%) women developed wound infection and 66 (24.6%)developed wound breakdown; however, only one woman (0.4%)developed a rectoperineal fistula.The natural history of rectoperineal fistula after obstetricinjuries has not been well-clarified; however, it might developas an epithelialized track that connects the perineal skin andthe rectum when a perianal abscess ruptures or is drained.As tight adhesion of the vaginal wall might block rectovaginalfistulation, the abscess might proceed toward the perineal skin,previously injured by an obstetric laceration or episiotomy. Thetiming of the occurrence of wound problems after obstetricinjury varied among several reports. The rectoperineal fistulain the case of Lewicky-Gaupp et al. was presented and treatedwithin 2 weeks of delivery (10). Stock et al. showed that thetiming of readmission of wound complications after obstetricanal sphincter injury was 9 weeks after delivery at the latest(11). On the other hand, Barranger et al. presented three casesof anal fistula as a late complication of mediolateral episiotomy,which had been diagnosed 5, 9, and 24 months after delivery,respectively (12). The early timing of fistulation was caused bybacteria-contaminated at vaginal delivery; however, it must beFrontiers in Surgery | www.frontiersin.org 3 June 2021 | Volume 8 | Article 637719Ohara et al. Rectoperineal Fistula After Obstetric LacerationFIGURE 3 | Surgical procedure of rectoperineal fistula repair. (A) Identification of rectoperineal fistula with a probe through the fistula tract. (B) Transverse perinealincision and dissection of rectovaginal septum exposing fistula tract. (C) Fistula tract is completely excised from the rectovaginal septum. Small box, macroscopicfeature of fistula tract (2 × 1 cm). (D) Layered closure of rectum, perineal body, vagina, and skin are performed, respectively. The skin was closed along verticaldirection (dotted arrow).noted that perianal abscess could be produced even at a later timeafter vaginal delivery.A rectoperineal fistula should be repaired in considerationto the rectovaginal fistula. Rectovaginal fistula can be treatedthrough abdominal, rectal, vaginal, perineal, trans-sphincteric,or trans-sacral approaches (13). For simple rectoperineal fistula,as in our case, a rectal or perineal approach followed by layerclosure might be appropriate. Our perineal approach could easilyrecognize and dissect the fistulous tract, avoiding sphinctericinjury. Due to the dissection of the rectovaginal septum, theposterior vaginal and anterior rectal wall are widely mobilized.Therefore, the layered closure of the rectal wall, perinealbody, and vaginal wall could tightly construct the perineumwithout tissue tension. Wiskind and Thompson reported thatall patients who underwent rectoperineal repair had satisfactoryrecovery with no recurrence in a series of 21 patients withrectovaginal fistula (14). Although the advancement flap throughthe rectal approach might be a procedure for rectoperinealfistula, it was not practiced in our case in which slight andfragile tissues remained between the vagina and rectum dueto the primary obstetric injury. A thick perineal cushion wasnecessary to prevent recurrence or anal incontinence, whichcould not be created with an advancement flap. Hull et al. alsoreported that 78% of patients who underwent rectoperineal repairFrontiers in Surgery | www.frontiersin.org 4 June 2021 | Volume 8 | Article 637719Ohara et al. Rectoperineal Fistula After Obstetric Lacerationafter obstetric injury was successfully repaired, providing betteroutcomes than the rectal advancement flap method (15).CONCLUSIONSevere obstetric perineal laceration can result in a vaginal oranorectal fistula even several months after primary repair, and thefistula may infrequently be opened at the perineal skin. Surgeryfor rectoperineal fistula requires complete fistulous tract excisionunder the distorted anatomy of the perineum and robust perinealreconstruction preventing recurrence and anal dysfunction.DATA AVAILABILITY STATEMENTThe raw data supporting the conclusions of this article will bemade available by the authors, without undue reservation.ETHICS STATEMENTThe studies involving human participants were reviewed andapproved by University of Tsukuba. The patients/participantsprovided their written informed consent to participate inthis study.AUTHOR CONTRIBUTIONSYOh, TE, and YOw performed surgical operation. KH, YA,KO, MD, KT, OS, KF, and SH planned and approved thesurgical treatments. RO, MO-Y, and HH managed the obstetrictreatment. JK supported the illustration in this article. YOhand TO were major contributors in writing the manuscript. Allauthors substantially contributed to the manuscript and read andapproved the final manuscript.REFERENCES1. Smith LA, Price N, Simonite V, Burns EE. Incidence of and risk factorsfor perineal trauma: a prospective observational study. BMC PregnancyChildbirth. (2013) 13:59. doi: 10.1186/1471-2393-13-592. Rogers RG, Leeman LM, Borders N, Qualls C, Fullilove AM, Teaf D, et al.Contribution of the second stage of labour to pelvic floor dysfunction:a prospective cohort comparison of nulliparous women. BJOG. (2014)121:1145–53; discussion 54. doi: 10.1111/1471-0528.125713. Practice Bulletin No. 165: Prevention and Management of ObstetricLacerations at Vaginal Delivery. Obstet Gynecol. (2016) 128:e115.doi: 10.1097/AOG.00000000000015234. Landy HJ, Laughon SK, Bailit JL, Kominiarek MA, Gonzalez-QuinteroVH, Ramirez M, et al. Characteristics associated with severe perineal andcervical lacerations during vaginal delivery. Obstet Gynecol. (2011) 117:627-35. doi: 10.1097/AOG.0b013e31820afaf25. Delancey JO, Toglia MR, Perucchini D. Internal and external anal sphincteranatomy as it relates to midline obstetric lacerations. Obstet Gynecol. (1997)90:924–7. doi: 10.1016/S0029-7844(97)00472-96. Goldaber KG, Wendel PJ, McIntire DD, Wendel GD, Jr. Postpartum perinealmorbidity after fourth-degree perineal repair. Am J Obstet Gynecol. (1993)168:489–93. doi: 10.1016/0002-9378(93)90478-27. Venkatesh KS, Ramanujam PS, Larson DM, Haywood MA. Anorectalcomplications of vaginal delivery. Dis Colon Rectum. (1989) 32:1039–41.doi: 10.1007/BF025538778. Handa VL, Danielsen BH, Gilbert WM. Obstetric anal sphincter lacerations.Obstet Gynecol. (2001) 98:225-30. doi: 10.1097/00006250-200108000-000089. Williams MK, Chames MC. Risk factors for the breakdown of perineallaceration repair after vaginal delivery. Am J Obstet Gynecol. (2006) 195:755-9.doi: 10.1016/j.ajog.2006.06.08510. Lewicky-Gaupp C, Leader-Cramer A, Johnson LL, Kenton K, GossettDR. Wound complications after obstetric anal sphincter injuries.Obstet Gynecol. (2015) 125:1088–93. doi: 10.1097/AOG.000000000000083311. Stock L, Basham E, Gossett DR, Lewicky-Gaupp C. Factors associatedwith wound complications in women with obstetric anal sphincterinjuries (OASIS). Am J Obstet Gynecol. (2013) 208:327.e1–6.doi: 10.1016/j.ajog.2012.12.02512. Barranger E, Haddad B, Paniel BJ. Fistula in ano as a rare complication ofmediolateral episiotomy: report of three cases. Am J Obstet Gynecol. (2000)182:733–4. doi: 10.1067/mob.2000.10296013. Gordon PH. Rectovaginal fistula. Principles and Practice of Surgery for theColon, Rectum, and Anus. 3rd ed. New York, NY: Informa Healthcare (2006).p. 333–52.14. Wiskind AK, Thompson JD. Transverse transperineal repair of rectovaginalfistulas in the lower vagina. Am J Obstet Gynecol. (1992) 167:694–9.doi: 10.1016/S0002-9378(11)91573-715. Hull TL, El-Gazzaz G, Gurland B, Church J, Zutshi M. Surgeons shouldnot hesitate to perform episioproctotomy for rectovaginal fistula secondaryto cryptoglandular or obstetrical origin. Dis Colon Rectum. (2011) 54:54–9.doi: 10.1097/01.dcr.0000388926.29548.36Conflict of Interest: The authors declare that the research was conducted in theabsence of any commercial or financial relationships that could be construed as apotential conflict of interest.Copyright © 2021 Ohara, Enomoto, Owada, Hisakura, Akashi, Ogawa, Doi,Takahashi, Shimomura, Furuya, Kim, Hashimoto, Ohara, Obata-Yasuoka, Hamadaand Oda. This is an open-access article distributed under the terms of the CreativeCommons Attribution License (CC BY). The use, distribution or reproduction inother forums is permitted, provided the original author(s) and the copyright owner(s)are credited and that the original publication in this journal is cited, in accordancewith accepted academic practice. No use, distribution or reproduction is permittedwhich does not comply with these terms.Frontiers in Surgery | www.frontiersin.org 5 June 2021 | Volume 8 | Article 637719

Rectoperineal Fistula Presented 5 Months After Repair of Severe Obstetric Perineal Laceration: A Case Report

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Rectoperineal Fistula Presented 5 Months After Repair of Severe Obstetric Perineal Laceration: A Case Report

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