Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion M. F. Aslam, J. Krashin & M. A. Denman To cite this article: M. F. Aslam, J. Krashin & M. A. Denman (2014) Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion, Journal of Obstetrics and Gynaecology, 34:6, 542-542, DOI: 10.3109/01443615.2014.914477 To link to this article: http://dx.doi.org/10.3109/01443615.2014.914477
Published online: 15 May 2014.
Submit your article to this journal
Article views: 34
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ijog20 Download by: [Monash University Library]
Date: 01 April 2016, At: 21:15
Downloaded by [Monash University Library] at 21:15 01 April 2016
542
Gynaecology Case Reports
or from metaplasia of the pelvic Müllerian cells (Khan et al. 2002). Most cases reported in literature have appeared after caesarean sections. It is believed that scar endometriosis results from iatrogenic inoculation of endometrial cells into the fascia or subcutaneous tissues during invasive abdominopelvic procedures. Therefore, it is strongly recommended that before closure, the abdominal wound is thoroughly cleaned and irrigated vigorously with saline (Teng et al. 2008). There is insufficient information in the literature about the optimal management of uterine or extragenital ESS. As in other sarcomas, surgery is the most effective treatment for ESS. The efficacy of adjuvant therapy has not been proven (Puliyath and Nair 2012). In this paper, we have presented a rare case of malignancy in extragenital endometriosis. The case demonstrates that the endometrial stromal sarcoma might originate from a pre-existing endometriosis in the absence of any primary foci in the uterus or ovaries. Malignant transformation of endometriosis should be considered in the differential diagnosis of any new pelvic lesion in a patient with a history of endometriosis. Clinicians should keep in mind that ESS derived from scar endometriosis might be seen in the abdominal wall. To the best of our knowledge, this is the first case of endometrial stromal sarcoma arising from rectus abdominis muscle described in the literature. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Brooks JJ, Wheeler JE. 1977. Malignancy arising in extragonadal endometriosis: a case report and summary of the world literature. Cancer 40:3065–3073. Higashiura Y, Kajihara H, Shigetomi H et al. 2012. Identification of multiple pathways involved in the malignant transformation of endometriosis. Oncology Letters 4:3–9. Jung SI, Shin SS, Choi C et al. 2009. Endometrial stromal sarcoma presenting as prevesical mass mimicking urachal tumor. Journal of Korean Medical Science 24:529–531. Khan AW, Craig M, Jarmulowicz M et al. 2002. Liver tumours due to endometriosis and endometrial stromal sarcoma. HPB (Oxford) 4:43–45. Lauslahti K. 1972. Malignant external endometriosis. Acta Pathologica Microbiologica Scandinavica 223:98–102. Puliyath G, Nair MK. 2012. Endometrial stromal sarcoma: A review of the literature. Indian Journal of Medical and Paediatric Oncology 33:1–6. Sampson J. 1925. Endometrial carcinoma of the ovary, arising in endometrial tissue in that organ. Archives of Surgery 10:1–72. Sofoudis C, Kalampokas T, Grigoriadis C et al. 2012. Endometrial stromal sarcoma in a 29-year-old patient. Case report and review of the literature. European Journal of Gynaecological Oncology 33:328–330. Teng CC, Yang HM, Chen KF et al. 2008. Abdominal wall endometriosis: An overlooked but possibly preventable complication. Taiwanese Journal of Obstetrics and Gynecology 47:42–48. Veda P, Srinivasaiah M. 2010. Incisional endometriosis: diagnosed by fine needle aspiration cytology. Journal of Laboratory Physicians 2:117–120.
Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion M. F. Aslam, J. Krashin & M. A. Denman From the Division of Urogynecology and Reconstructive Pelvic Surgery, Department of Obstetrics and Gynecology, Oregon Health and Science University, Portland, Oregon, USA DOI: 10.3109/01443615.2014.914477 Correspondence: M. F. Aslam, Department of Obstetrics and Gynecology, Mail Code L466, OHSU, SW Sam Jackson Park Road, Portland, Oregon 97239, USA. E-mail: [email protected]
Case report A 48-year-old patient was referred to us at the Department of Female Pelvic Medicine and Reconstructive Surgery at the University Hospital, due to continuous urinary leakage for the past 8 months. At that time (8 months earlier), she had a transobturator tape (TOT) sling inserted for stress incontinence of urine, that was not proven by preoperative urodynamic assessment, in another hospital. The urethra was injured at the time and repaired in a single layer with interrupted chromic catgut sutures. Check cystoscopy was carried out at the time. The bladder was drained with Foley’s catheter for 14 days afterwards. However, she suffered from uncontrollable leakage of urine following removal of the catheter. Urethro-vaginal fistula was confirmed on examination, with methylene blue test, as well as voiding cystourethrography. The patient was counselled about the findings and agreed to examination under anaesthesia, with a view towards excision of the tape and repair of the fistula. Initial cystoscopy revealed a normal bladder with transurethral erosion of clear mesh material at the level of the bladder neck and a 1 cm urethral defect distal to this. She underwent transvaginal excision of the tape at the level of the bladder neck and repair of the urethro-vaginal fistula. The repair necessitated re-attachment of the inferior aspect of the urethra to the bladder base following separation of these structures with mesh removal. The defect was repaired in a two-layer fashion with 4-0 Vicryl suture. A total of 300 ml of diluted methylene blue was instilled into the bladder, confirming watertight repair with no leak seen after catheter removal and manual occlusion of the external urethral meatus. A bulbocavernosus fat pad (Martius flap) was developed and brought into the field to cover the defect. A 2 ⫻ 3 cm piece of small intestine submucosa (SIS) graft was placed, as there was limited tissue to bring into the suture area for reinforcement. The graft was used to bridge the vaginal mucosa. The patient was discharged home with a transurethral Foley’s catheter. She was advised to stop smoking to facilitate wound healing. The Foley’s catheter was removed 3 weeks postoperatively, following a voiding cystourethrogram demonstrating no recurrent fistula. She did well for 4 weeks and then reported a return of large volume leakage. Examination demonstrated recurrent fistula at the bladder neck. The patient was counselled regarding the findings and underwent a second transvaginal, multilayered repair of a 3 cm bladder neck fistula, with the same techniques as used previously. Increased tissue mobilisation was achieved. Tissue mobilisation was achieved for at least 2 cm circumferentially around the fistula. The fistula edges were excised prior to closure. An additional midurethral approximation of the lateral edges of the pubococcygeus muscle was performed to facilitate bladder neck support, following watertight repair. A transurethral Foley’s catheter was again placed for 3 weeks and removed following a voiding cystourethrogram demonstrating no recurrence of the fistula. Postoperatively, she developed stress urinary incontinence, which responded to a transurethral bulking agent (Macroplastique®, Uroplasty) 4 months following her second repair. She was seen in follow-up 1 month following injection, with no complaints of urinary leakage.
Conclusion Synthetic midurethral slings are commonly performed for stress urinary incontinence. Recent studies have shown the transobturator method has fewer bladder complications or other adverse events compared with the retropubic approach, concluding that the transobturator approach is safe and effective for the treatment of stress incontinence due to fewer complications (Cheng and Liu 2012). The risk of urethral injury remains, regardless of approach. Fistula formation however, is exceedingly rare. Of the reported cases of urogenital fistula after synthetic slings for incontinence, none relate to the placement of tape after urethral injury, which is unique in our case (Morton and Hilton 2009). Our presented case combines two uncommon long-term complications: urethral mesh erosion and recurrent fistula. We posit the unusual combination is related to the urethral injury, repair and placement of foreign body over the repair site. Increased inflammation in a single layer repair
Downloaded by [Monash University Library] at 21:15 01 April 2016
Gynaecology Case Reports 543 risks breakdown of the suture line, with exposure to the underlying tissue or space. In this case, both the uses of chromic catgut in the initial repair, as well as the placement of polypropylene mesh, risk increasing the local inflammatory response (Kosan et al. 2008; Huber et al. 2012). This combination possibly compromised healing, resulting in long-term sequelae for the patient. The failure of the initial fistula repair may have been attributable to poor wound healing secondary to the patient’s tobacco use, increased inflammatory response from mesh removal, use of small intestine submucosa (SIS) graft or subclinical infection related to the transurethral mesh exposure. These are possible contributory factors. The subjectively improved condition of the tissue at the time of the second repair suggests a baseline inflammatory state associated with the mesh erosion contributing to surgical failure of the initial repair. We have no objective data at this time to support the use of biological mesh in repair of urethro-vaginal fistula. To our knowledge, there are no studies or case reports regarding placement of mesh after urethral injury. The American Urological Association recommends against placement of synthetic sling if intraoperative urethral injury occurs on the basis of panel consensus (Dmochowski et al. 2010). This case supports this position statement. We learn from this case report that if urethral injury occurs at the time of tape placement, we should defer the placement of tape. This is based on limited evidence and panel opinion. The repair of fistula should be delayed until the inflammation has subsided, to achieve adequate tissue placement. With the increased attention from consumers, regulatory bodies and the legal community directed at transvaginal mesh and its complications, optimising patient outcomes and the avoidance of long-term injury should remain of utmost concern to the surgeon. Women undergoing midurethral sling procedures for stress urinary incontinence should be counselled of these serious but rare complications. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Cheng D, Liu C. 2012. Tension-free vaginal tape-obturator in the treatment of stress urinary incontinence: a prospective study with five-year follow-up. European Journal of Obstetrics & Gynecology, and Reproductive Biology 161:228–231. Dmochowski RR, Blaivas JM, Gormley EA et al. 2010. Update of AUA Guideline on the Surgical Management of Female Stress Urinary Incontinence. Journal of Urology 183:1906–1914. Huber A, Boruch AV, Nieponice A et al. 2012. Histopathologic host response to polypropylene-based surgical mesh materials in a rat abdominal wall defect model. Journal of Biomedical Materials Research. Part B, Applied Biomaterials 100:709–717. Kosan M, Gonulalan U, Ozturk B et al. 2008. Tissue reactions of suture materials (polyglactine 910, chromed catgut and polydioxanone) on rat bladder wall and their role in bladder stone formation. Urological Research 36:43–49. Morton HC, Hilton P. 2009. Urethral injury associated with minimally invasive mid-urethral sling procedures for the treatment of stress urinary incontinence: a case series and systemic literature search. British Journal of Obstetrics and Gynaecology 116:1120–1126.
Introduction Müllerian malformations include a broad range of anomalies, resulting from the incomplete formation and/or differentiation of Müllerian ducts. Uterus didelphys with obstructed hemivagina is the result of a lateral non-fusion of the Müllerian ducts with asymmetric obstruction (Golan et al. 1999). Uterus didelphys arises from midline fusion arrest of the Müllerian ducts, in either a complete or incomplete form, and it comprises 11% of uterine malformations. The complete form has two hemiuteri and two endocervical canals, with cervices fused at the lower uterine segment. The vagina may be single or double, in which case a longitudinal septum is present that extends either completely or partially from the cervices to the introitus (Zurawin et al. 2004). Renal agenesis associated with uterus didelphys is more frequent than with any other type of Müllerian anomaly. The reported incidence of renal anomalies in this group of patients is 20% (Tanaka et al. 1998). Patients are usually asymptomatic unless there is an obstruction, in which case haematometrocolpos, haematometra and haematosalpinx may develop. In hemivaginal obstruction, the most common presenting symptoms are onset of dysmenorrhoea within the first years following menarche and progressive pelvic pain. A unilateral pelvic mass may be present (Vercellini et al. 2007). An obstructed unilateral vagina is an indication for vaginal septum resection. Surgery is necessary to preserve reproductive capacity and to prevent uterine and tubal impairment. Unless the obstruction is readily removed after diagnosis, retrograde menstruation continues and haematometra, haematosalpinx, endometriosis and pelvic adhesions may develop.
Case report A 16-year-old girl with a dysmenorrhoea complaint since menarche and progressive pelvic pain was referred to our institution. She had a history of left nephrectomy for a non-functional pelvic kidney at the age of 9. At physical examination, she had an intact annular hymen but no palpable abdominal masses or tenderness. Transabdominal ultrasound and magnetic resonance imaging (MRI) revealed a complete uterus didelphys with a double vagina that was distended on the left side with a complete septum (Figure 1). The patient was medicated with a combined oral contraceptive (COC) pill to trigger withdrawal bleeding and subjected to diagnostic laparoscopy and vaginal septum removal, allowing left hemi-vagina opening. During the procedure, two uterine formations suggestive of didelphys uterus were observed at laparoscopy. Right tube permeability was confirmed laparoscopically after chromopertubation. Peroperative hysterosalpingography showed only a right uterine cavity with respective tubal permeability.
Finding the obstructed hemivagina in uterus didelphys D. M. da Silva Coelho1, R. F. Sousa Santos1,2 & M. L. Graça Fernandes1 1Department of Obstetrics and Gynaecology, Centro Hospitalar do Alto Ave, Guimarães and 2CINTESIS, Faculty of Medicine, University of Porto, Porto, Portugal
DOI: 10.3109/01443615.2014.915292 Correspondence: D. M. da Silva Coelho, Rua dos Cutileiros, Creixomil, 4835-044 Guimarães, Portugal. E-mail: [email protected]
Figure 1. Pelvic magnetic resonance imaging revealing complete didelphys uterus with double vagina, distended on the right side with a complete septum.
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion M. F. Aslam, J. Krashin & M. A. Denman To cite this article: M. F. Aslam, J. Krashin & M. A. Denman (2014) Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion, Journal of Obstetrics and Gynaecology, 34:6, 542-542, DOI: 10.3109/01443615.2014.914477 To link to this article: http://dx.doi.org/10.3109/01443615.2014.914477
Published online: 15 May 2014.
Submit your article to this journal
Article views: 34
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ijog20 Download by: [Monash University Library]
Date: 01 April 2016, At: 21:15
Downloaded by [Monash University Library] at 21:15 01 April 2016
542
Gynaecology Case Reports
or from metaplasia of the pelvic Müllerian cells (Khan et al. 2002). Most cases reported in literature have appeared after caesarean sections. It is believed that scar endometriosis results from iatrogenic inoculation of endometrial cells into the fascia or subcutaneous tissues during invasive abdominopelvic procedures. Therefore, it is strongly recommended that before closure, the abdominal wound is thoroughly cleaned and irrigated vigorously with saline (Teng et al. 2008). There is insufficient information in the literature about the optimal management of uterine or extragenital ESS. As in other sarcomas, surgery is the most effective treatment for ESS. The efficacy of adjuvant therapy has not been proven (Puliyath and Nair 2012). In this paper, we have presented a rare case of malignancy in extragenital endometriosis. The case demonstrates that the endometrial stromal sarcoma might originate from a pre-existing endometriosis in the absence of any primary foci in the uterus or ovaries. Malignant transformation of endometriosis should be considered in the differential diagnosis of any new pelvic lesion in a patient with a history of endometriosis. Clinicians should keep in mind that ESS derived from scar endometriosis might be seen in the abdominal wall. To the best of our knowledge, this is the first case of endometrial stromal sarcoma arising from rectus abdominis muscle described in the literature. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Brooks JJ, Wheeler JE. 1977. Malignancy arising in extragonadal endometriosis: a case report and summary of the world literature. Cancer 40:3065–3073. Higashiura Y, Kajihara H, Shigetomi H et al. 2012. Identification of multiple pathways involved in the malignant transformation of endometriosis. Oncology Letters 4:3–9. Jung SI, Shin SS, Choi C et al. 2009. Endometrial stromal sarcoma presenting as prevesical mass mimicking urachal tumor. Journal of Korean Medical Science 24:529–531. Khan AW, Craig M, Jarmulowicz M et al. 2002. Liver tumours due to endometriosis and endometrial stromal sarcoma. HPB (Oxford) 4:43–45. Lauslahti K. 1972. Malignant external endometriosis. Acta Pathologica Microbiologica Scandinavica 223:98–102. Puliyath G, Nair MK. 2012. Endometrial stromal sarcoma: A review of the literature. Indian Journal of Medical and Paediatric Oncology 33:1–6. Sampson J. 1925. Endometrial carcinoma of the ovary, arising in endometrial tissue in that organ. Archives of Surgery 10:1–72. Sofoudis C, Kalampokas T, Grigoriadis C et al. 2012. Endometrial stromal sarcoma in a 29-year-old patient. Case report and review of the literature. European Journal of Gynaecological Oncology 33:328–330. Teng CC, Yang HM, Chen KF et al. 2008. Abdominal wall endometriosis: An overlooked but possibly preventable complication. Taiwanese Journal of Obstetrics and Gynecology 47:42–48. Veda P, Srinivasaiah M. 2010. Incisional endometriosis: diagnosed by fine needle aspiration cytology. Journal of Laboratory Physicians 2:117–120.
Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion M. F. Aslam, J. Krashin & M. A. Denman From the Division of Urogynecology and Reconstructive Pelvic Surgery, Department of Obstetrics and Gynecology, Oregon Health and Science University, Portland, Oregon, USA DOI: 10.3109/01443615.2014.914477 Correspondence: M. F. Aslam, Department of Obstetrics and Gynecology, Mail Code L466, OHSU, SW Sam Jackson Park Road, Portland, Oregon 97239, USA. E-mail: [email protected]
Case report A 48-year-old patient was referred to us at the Department of Female Pelvic Medicine and Reconstructive Surgery at the University Hospital, due to continuous urinary leakage for the past 8 months. At that time (8 months earlier), she had a transobturator tape (TOT) sling inserted for stress incontinence of urine, that was not proven by preoperative urodynamic assessment, in another hospital. The urethra was injured at the time and repaired in a single layer with interrupted chromic catgut sutures. Check cystoscopy was carried out at the time. The bladder was drained with Foley’s catheter for 14 days afterwards. However, she suffered from uncontrollable leakage of urine following removal of the catheter. Urethro-vaginal fistula was confirmed on examination, with methylene blue test, as well as voiding cystourethrography. The patient was counselled about the findings and agreed to examination under anaesthesia, with a view towards excision of the tape and repair of the fistula. Initial cystoscopy revealed a normal bladder with transurethral erosion of clear mesh material at the level of the bladder neck and a 1 cm urethral defect distal to this. She underwent transvaginal excision of the tape at the level of the bladder neck and repair of the urethro-vaginal fistula. The repair necessitated re-attachment of the inferior aspect of the urethra to the bladder base following separation of these structures with mesh removal. The defect was repaired in a two-layer fashion with 4-0 Vicryl suture. A total of 300 ml of diluted methylene blue was instilled into the bladder, confirming watertight repair with no leak seen after catheter removal and manual occlusion of the external urethral meatus. A bulbocavernosus fat pad (Martius flap) was developed and brought into the field to cover the defect. A 2 ⫻ 3 cm piece of small intestine submucosa (SIS) graft was placed, as there was limited tissue to bring into the suture area for reinforcement. The graft was used to bridge the vaginal mucosa. The patient was discharged home with a transurethral Foley’s catheter. She was advised to stop smoking to facilitate wound healing. The Foley’s catheter was removed 3 weeks postoperatively, following a voiding cystourethrogram demonstrating no recurrent fistula. She did well for 4 weeks and then reported a return of large volume leakage. Examination demonstrated recurrent fistula at the bladder neck. The patient was counselled regarding the findings and underwent a second transvaginal, multilayered repair of a 3 cm bladder neck fistula, with the same techniques as used previously. Increased tissue mobilisation was achieved. Tissue mobilisation was achieved for at least 2 cm circumferentially around the fistula. The fistula edges were excised prior to closure. An additional midurethral approximation of the lateral edges of the pubococcygeus muscle was performed to facilitate bladder neck support, following watertight repair. A transurethral Foley’s catheter was again placed for 3 weeks and removed following a voiding cystourethrogram demonstrating no recurrence of the fistula. Postoperatively, she developed stress urinary incontinence, which responded to a transurethral bulking agent (Macroplastique®, Uroplasty) 4 months following her second repair. She was seen in follow-up 1 month following injection, with no complaints of urinary leakage.
Conclusion Synthetic midurethral slings are commonly performed for stress urinary incontinence. Recent studies have shown the transobturator method has fewer bladder complications or other adverse events compared with the retropubic approach, concluding that the transobturator approach is safe and effective for the treatment of stress incontinence due to fewer complications (Cheng and Liu 2012). The risk of urethral injury remains, regardless of approach. Fistula formation however, is exceedingly rare. Of the reported cases of urogenital fistula after synthetic slings for incontinence, none relate to the placement of tape after urethral injury, which is unique in our case (Morton and Hilton 2009). Our presented case combines two uncommon long-term complications: urethral mesh erosion and recurrent fistula. We posit the unusual combination is related to the urethral injury, repair and placement of foreign body over the repair site. Increased inflammation in a single layer repair
Downloaded by [Monash University Library] at 21:15 01 April 2016
Gynaecology Case Reports 543 risks breakdown of the suture line, with exposure to the underlying tissue or space. In this case, both the uses of chromic catgut in the initial repair, as well as the placement of polypropylene mesh, risk increasing the local inflammatory response (Kosan et al. 2008; Huber et al. 2012). This combination possibly compromised healing, resulting in long-term sequelae for the patient. The failure of the initial fistula repair may have been attributable to poor wound healing secondary to the patient’s tobacco use, increased inflammatory response from mesh removal, use of small intestine submucosa (SIS) graft or subclinical infection related to the transurethral mesh exposure. These are possible contributory factors. The subjectively improved condition of the tissue at the time of the second repair suggests a baseline inflammatory state associated with the mesh erosion contributing to surgical failure of the initial repair. We have no objective data at this time to support the use of biological mesh in repair of urethro-vaginal fistula. To our knowledge, there are no studies or case reports regarding placement of mesh after urethral injury. The American Urological Association recommends against placement of synthetic sling if intraoperative urethral injury occurs on the basis of panel consensus (Dmochowski et al. 2010). This case supports this position statement. We learn from this case report that if urethral injury occurs at the time of tape placement, we should defer the placement of tape. This is based on limited evidence and panel opinion. The repair of fistula should be delayed until the inflammation has subsided, to achieve adequate tissue placement. With the increased attention from consumers, regulatory bodies and the legal community directed at transvaginal mesh and its complications, optimising patient outcomes and the avoidance of long-term injury should remain of utmost concern to the surgeon. Women undergoing midurethral sling procedures for stress urinary incontinence should be counselled of these serious but rare complications. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Cheng D, Liu C. 2012. Tension-free vaginal tape-obturator in the treatment of stress urinary incontinence: a prospective study with five-year follow-up. European Journal of Obstetrics & Gynecology, and Reproductive Biology 161:228–231. Dmochowski RR, Blaivas JM, Gormley EA et al. 2010. Update of AUA Guideline on the Surgical Management of Female Stress Urinary Incontinence. Journal of Urology 183:1906–1914. Huber A, Boruch AV, Nieponice A et al. 2012. Histopathologic host response to polypropylene-based surgical mesh materials in a rat abdominal wall defect model. Journal of Biomedical Materials Research. Part B, Applied Biomaterials 100:709–717. Kosan M, Gonulalan U, Ozturk B et al. 2008. Tissue reactions of suture materials (polyglactine 910, chromed catgut and polydioxanone) on rat bladder wall and their role in bladder stone formation. Urological Research 36:43–49. Morton HC, Hilton P. 2009. Urethral injury associated with minimally invasive mid-urethral sling procedures for the treatment of stress urinary incontinence: a case series and systemic literature search. British Journal of Obstetrics and Gynaecology 116:1120–1126.
Introduction Müllerian malformations include a broad range of anomalies, resulting from the incomplete formation and/or differentiation of Müllerian ducts. Uterus didelphys with obstructed hemivagina is the result of a lateral non-fusion of the Müllerian ducts with asymmetric obstruction (Golan et al. 1999). Uterus didelphys arises from midline fusion arrest of the Müllerian ducts, in either a complete or incomplete form, and it comprises 11% of uterine malformations. The complete form has two hemiuteri and two endocervical canals, with cervices fused at the lower uterine segment. The vagina may be single or double, in which case a longitudinal septum is present that extends either completely or partially from the cervices to the introitus (Zurawin et al. 2004). Renal agenesis associated with uterus didelphys is more frequent than with any other type of Müllerian anomaly. The reported incidence of renal anomalies in this group of patients is 20% (Tanaka et al. 1998). Patients are usually asymptomatic unless there is an obstruction, in which case haematometrocolpos, haematometra and haematosalpinx may develop. In hemivaginal obstruction, the most common presenting symptoms are onset of dysmenorrhoea within the first years following menarche and progressive pelvic pain. A unilateral pelvic mass may be present (Vercellini et al. 2007). An obstructed unilateral vagina is an indication for vaginal septum resection. Surgery is necessary to preserve reproductive capacity and to prevent uterine and tubal impairment. Unless the obstruction is readily removed after diagnosis, retrograde menstruation continues and haematometra, haematosalpinx, endometriosis and pelvic adhesions may develop.
Case report A 16-year-old girl with a dysmenorrhoea complaint since menarche and progressive pelvic pain was referred to our institution. She had a history of left nephrectomy for a non-functional pelvic kidney at the age of 9. At physical examination, she had an intact annular hymen but no palpable abdominal masses or tenderness. Transabdominal ultrasound and magnetic resonance imaging (MRI) revealed a complete uterus didelphys with a double vagina that was distended on the left side with a complete septum (Figure 1). The patient was medicated with a combined oral contraceptive (COC) pill to trigger withdrawal bleeding and subjected to diagnostic laparoscopy and vaginal septum removal, allowing left hemi-vagina opening. During the procedure, two uterine formations suggestive of didelphys uterus were observed at laparoscopy. Right tube permeability was confirmed laparoscopically after chromopertubation. Peroperative hysterosalpingography showed only a right uterine cavity with respective tubal permeability.
Finding the obstructed hemivagina in uterus didelphys D. M. da Silva Coelho1, R. F. Sousa Santos1,2 & M. L. Graça Fernandes1 1Department of Obstetrics and Gynaecology, Centro Hospitalar do Alto Ave, Guimarães and 2CINTESIS, Faculty of Medicine, University of Porto, Porto, Portugal
DOI: 10.3109/01443615.2014.915292 Correspondence: D. M. da Silva Coelho, Rua dos Cutileiros, Creixomil, 4835-044 Guimarães, Portugal. E-mail: [email protected]
Figure 1. Pelvic magnetic resonance imaging revealing complete didelphys uterus with double vagina, distended on the right side with a complete septum.
