Tech Coloproctol DOI 10.1007/s10151-014-1176-2
TECHNICAL NOTE
Ultrasonographic evaluation of anal endometriosis: report of four cases M. Kołodziejczak • I. Sudoł-Szopin´ska • G. A. Santoro • K. Bielecki • A. Wia˛czek
Received: 1 January 2014 / Accepted: 30 May 2014 Ó Springer-Verlag Italia Srl 2014
Abstract Background The presence of endometriosis in the anal canal and perianal tissues is rare and difficult to suspect at clinical examination. We report our experience with preoperative ultrasound evaluation of four cases of anal endometriosis. Methods Four patients were evaluated by transperineal and high-resolution three-dimensional endoanal ultrasonography. Results In 3 of 4 women, the lesions involved old episiotomy scars. Anal endometriosis appeared as hypoechoic cystic lesions with areas of microcalcification, not well delimited and highly vascularized. The lesions either involved the anal sphincter (n = 2, one within the rectovaginal septum) or were localized superficially in the ischiorectal space (n = 2). Surgery and pathologic exam confirmed the ultrasonographic findings. Conclusions Ultrasonographic findings of anal endometriosis are characteristics and may allow accurate preoperative staging of the disease.
M. Kołodziejczak K. Bielecki A. Wia˛czek Department of General Surgery, Sub-Department of Proctology, Hospital at Solec, 00-382 Warsaw, Poland I. Sudoł-Szopin´ska Department of Radiology, Institute of Rheumatology, Warsaw, Poland I. Sudoł-Szopin´ska Department of Diagnostic Imaging, Warsaw Medical University, Spartan´ska 1 St. 8, 02-637 Warsaw, Poland G. A. Santoro (&) Pelvic Floor Unit, I Department of Surgery, Regional Hospital, Piazzale Ospedale 1, 31100 Treviso, Italy e-mail: [email protected]
Keywords Anal sphincters Endoanal ultrasonography Endometriosis Transperineal ultrasonography
Introduction Endometriosis is a chronic disease, affecting 10–15 % of women of reproductive age, which characterized by the ectopic localization of the hormonally active uterine endometrium [1]. It has an unclear and complex pathophysiology, including different risk factors: history of gynecological surgery, high estrogen levels, early menarche, short menstrual cycles (\27 days), menstrual bleeding for [7 days, and immune system disorders [2]. According to the implantation theory, the endometrial cells travel through the fallopian tubes or may also reach remote organs via blood and lymphatic vessels [3]. Typical localizations of endometriosis are the uterosacral ligaments and ovaries (approximately 60 %), the pouch of Douglas (25 %), the vesicouterine space (in the form of tarry cysts) (20 %), the round ligament of the uterus (11 %), and the bowel (7 %) [4]. Rarely, endometriotic foci can be found in the anal canal and perianal tissues [5–8]. Symptoms of endometriosis are pain in the lower pelvis or in the perineum (particularly during the menstrual cycle), menstrual disorders, pain during sexual intercourse, and infertility. Preoperative imaging of anal endometriosis in order to determine whether the foci involve the anal sphincters or are limited to the perianal space is fundamental for planning adequate surgical treatment. In this paper, we report our experience with four cases assessed by transperineal (TPUS) and high-resolution three-dimensional (3D) endoanal ultrasound (EAUS).
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Imaging techniques Endoanal ultrasonography (EAUS) EAUS was performed with the patient placed in the left lateral decubitus, using a 360° rotating, mechanical, multifrequencies (frequency range 6–16 MHz) transducer with automatic, computed-controlled (300 transaxial images, over a distance of 60 mm in 60 s), high-resolution 3D acquisition (type 2050, BK Medical Analogic Corporation, Herlev, Denmark) [9, 10]. In the axial plane, the anal canal was divided into three levels of assessment referring to the following anatomic structures: (1) upper level: the hyperechoic sling of the puborectalis muscle (PR) and the complete hypoechoic ring of the internal anal sphincter (IAS); (2) middle level: the superficial, mixed echogenicity ring of the external anal sphincter (EAS), the conjoined longitudinal layer, the IAS, and the transverse superficial perinei muscles; and (3) lower level: the subcutaneous EAS. The 3D reconstruction was useful in assessing the anatomic characteristics of the anal canal in the longitudinal and coronal planes (MPR = multiplanar reconstructions).
proctological disease were reported. Her obstetrical history was characterized by a vaginal delivery 11 years before. On rectal examination, a mobile and painful nodule of approximately 4 cm was found in the posterior, right lateral quadrant of the anal canal, at 1 cm from the anal verge. The resting and squeeze pressures of the sphincter muscles were normal. Proctoscopy revealed a normal mucosal surface. On TPUS, a subcutaneous, hypoechoic, and highly vascularized lesion was revealed. It was not well circumscribed, and its dimensions were 2.1 9 3.5 cm (Fig. 1a). On EUAS, an hypoechoic lesion was visualized in the perianal tissue, adjacent to but noninvading the outer border of the subcutaneous EAS (Fig. 1b). Based on the ultrasound (US) findings that confirmed the absence of a chronic abscess, the patient underwent surgery. A hard mass of 3 9 5 cm, localized in the right perianal space, adjacent to the anal sphincter, was excised. Histological analysis of the specimen revealed a focus of endometriosis. The patient was given hormonal therapy for 10 months after surgery. At the clinical and US follow-up at 2 years, no recurrence was found. Case 2
We report four cases of anal endometriosis assessed with both 3D-EAUS and TPUS (Table 1). The protocol was approved by the Medical University’s review board, and all participants gave written informed consent.
A 42-year-old woman presented with an anovaginal fistula. She had had two vaginal deliveries with episiotomy. At clinical assessment, the vaginal orifice of the fistula was located at 1.5 cm from the vaginal verge and the anal orifice at 2 cm from the anal margin. Proctoscopy (up to 20 cm) confirmed the presence of the anal opening. The surface of the rectal mucosa was normal. On EAUS, both the anovaginal tract and the internal opening (IO) at the level of the anorectum were visualized. No collection was demonstrated (Fig. 2). At surgery, the fistula with its anal opening was excised and the defect was closed with an anocutaneous advancement flap. The vaginal opening was also sutured with a series of absorbable stitches. There were no postoperative complications. Histological analysis revealed the presence of fibrous tissue and chronic inflammation. Foci of endometriosis were found in the stroma. The patient, following gynecological consultation, received hormonal therapy. At 3-year follow-up, clinical examination and ultrasonographic assessment confirmed the healing of the fistula and the presence of a scar.
Case 1
Case 3
A 34-year-old woman was admitted to our department with suspected anal abscess. She complained of a painful, perianal mass, without purulent discharge or rectal bleeding, first noticed 12 months prior, ineffectively treated with antibiotics. No fever, trauma of the perineum, or any other
A 33-year-old woman with a 2 cm painful nodule localized in the episiotomy scar from her vaginal delivery 12 years prior was admitted to our department for surgical treatment. She did not complain of other symptoms. On TPUS, an hypoechoic, not well delimited, vascularized area
Transperineal ultrasonography (TPUS) Transperineal ultrasound was performed with the patient placed in dorsal lithotomy, with the hips flexed and abducted, using a 2D linear array, multifrequencies (frequency range 5–12 MHz) transducer (type 8811, BK Medical Analogic Corporation). The probe was positioned in the perineum, slightly anterior to the anus, and was rotated in order to provide sagittal, coronal, and oblique sectional imaging of the anal canal and perianal tissues. Power Doppler (PWD) option allowed evaluation of vascularity of the lesions [11, 12].
Case reports
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Tech Coloproctol Table 1 Clinical and ultrasonographic findings in patients with anal and perianal endometriosis Patient no.
History
Clinical examination
Transperineal ultrasonography
3D endoanal ultrasonography
Surgical treatment
1.
34-year-old woman with suspected anal abscess
Mobile and painful 4 cm nodule, in the posterior, right lateral quadrant of the anal canal, at 1 cm from the anal margin
Subcutaneous, 2.1 9 3.5 cm size, hypoechoic and highly vascularized lesion, not well circumscribed
Hypoechoic lesion in the perianal tissue, adjacent but noninvading the outer border of the subcutaneous EAS
Excision of a firm lesion, 3 9 5 cm, located in the right perianal space, adjacent to the anal sphincter
2.
42-year-old woman with an anovaginal fistula
Anovaginal fistula at 1.5 cm from the vaginal verge and at 2 cm from the anal margin
Subcutaneous fistulous tract in the perineal tissue
Anovaginal tract with visualization of the internal anal opening. No other collection. Regular EAS
Excision of the fistula with its internal anal opening; anocutaneous advancement flap; suture of the vaginal opening
3.
33-year-old woman with a 2 cm painful nodule located in the episiotomy scar
2 cm painful nodule in the episiotomy scar
Hypoechoic, not well delimited, vascularized area. 2 9 3 cm, with small cysts inside, located the anterior, right lateral quadrant of the anal canal
Hypoechoic lesion involving the anterior 1/3 of the superficial EAS, the adjacent intersphincteric space and the perineal tissue
Excision of a nodule, in the deeper part of the rectovaginal septum, with a small portion of the anterior EAS. Repair of the EAS with ‘‘end-toend’’ technique
4.
39-year-old woman with recurrence of endometriosis located in the episiotomy scar
4 cm nodule in the anterior and left lateral quadrants of the anal canal
Two hypoechoic nodules, 8 and 12 mm, not well delimited, with posterior wall enhancement, highly vascularized, with small cystic areas inside and microcalcifications
Hypoechoic lesion 4 9 2 cm in the anterior and left lateral quadrants of the anal canal, involving the ischiorectal fossa, the perineum and the lateral border of the EAS
Excision of two nodules, dissected from the fibers of the EAS
EAS external anal sphincter
2 9 3 cm with small cysts was detected in the anterior, right lateral quadrant of the anal canal. On 3D EAUS, the lesion involved the anterior 1/3 of the superficial EAS, the adjacent intersphincteric space, and the perineal tissue. No fistulous tracts were visualized (Fig. 3a). At surgery, the nodule, which penetrated into the deeper part of the rectovaginal septum, was completely excised with a small portion of the anterior EAS (Fig. 3b). There was no invasion of the rectal and vaginal walls. The anal sphincter was repaired with an ‘‘end-to-end’’ technique. The patient was discharged on the sixth postoperative day. Histological analysis demonstrated an endometriotic lesion. The patient received hormonal treatment. At 12-month follow-up, no recurrence was revealed. Case 4 A 39-year- old woman underwent excision of 2.5 cm focus of endometriosis in her episiotomy scar, 12 years after a vaginal delivery with episiotomy. At 6-month follow-up, she presented a 1 cm recurrence that was treated with hormonal therapy. However, at 1-year follow-up, the lesion had grown to approximately 4 cm.
On proctologic examination, a nodule of 4 cm was found in the anterior and left lateral quadrants of the anal sphincters. Proctoscopy up to 20 cm was normal. Transperineal US demonstrated two nodules, 8 and 12 mm in diameter. The lesions were hypoechoic with posterior wall enhancement, not well delimited, highly vascularized, with microcalcifications and small cystic areas (Fig. 4a, b). At EAUS, the lesion measured 4 9 2 cm and was localized in the anterior and left lateral quadrants of the anal canal. It involved the ischiorectal fossa, the perineum, and the lateral border of the subcutaneous and superficial parts of the EAS (Fig. 4c). At surgery, these two nodules were dissected from the fibers of the EAS and were completely excised. Histological analysis confirmed the recurrence of endometriotic foci. The patient was discharged on postoperative day 2.
Discussion The presence of endometriosis in the anal canal, in the ischiorectal fossa, and perianal tissues is rare and difficult to suspect at clinical examination. Detailed imaging is
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Fig. 2 Endoanal ultrasound showing the internal orifice (IO) of the anovaginal fistula in the anterior part of the middle level of the anal canal (EAS external anal sphincter)
Fig. 1 a Transperineal ultrasound shows an hypoechoic, highly vascularized, and not well-circumscribed lesion, 21 9 35 mm, at the right side of the anus. b At endoanal ultrasound, the lesion (L) appears in contact, but not invading, the subcutaneous part of the EAS
mandatory in order to exclude more common causes as perianal abscesses or anal tumors and for planning adequate management, and defining the size and position of the lesion as well as its relationship with the anal sphincter muscles. Magnetic resonance imaging (MRI) provides important information on the extension of endometriotic disease, helping to identify deep pelvic endometriosis in the retrocervical region, uterosacral and round ligaments, rectosigmoid, vagina, urinary tract, and other subperitoneal and extraperitoneal pelvic sites [13, 14]. Bazot et al. [15] reported that the sensitivity, specificity, positive and negative predictive values, and accuracy of MRI for deep pelvic endometriosis were 90.3, 91, 92, 89, and 90.6 %, respectively. However, EAUS has been defined the gold standard for assessment of the anal canal [16, 17]. 3D US with high spatial resolution allows multiplanar evaluation
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and good tissue characterization of the sphincter muscles, the perianal tissues, and the rectovaginal septum [9]. In addition, TPUS allows the assessment of the vascularity by using PWD. MRI and US should not be considered as alternative modalities, but can be combined in selected cases of deep infiltrating endometriosis, thereby facilitating accurate diagnosis and adequate treatment [18]. In our study, at TPUS, anal endometriosis was most frequently visualized as an hypoechoic or mixed echogenicity lesion, with areas of microcalcification or small cysts, not well delimited and highly vascularized. The relationship of the lesion with the anal sphincters and the rectal wall could be evaluated by EAUS. In Case 3, the lesion involved the EAS and these data were confirmed intraoperatively. The focus was excised, and the muscle was repaired. In Case 4, the lesion was in adjacent to but not invading the outer border of the EAS. At surgery, we could dissect the focus from the EAS, without removing muscle fibers. EAUS also helps in identifying anal sepsis or a fistula. In Case 2, an anovaginal fistula was demonstrated and subsequently treated by an anocutaneous advancement flap. Moreover, US is important in follow-up to confirm presence of scar tissue or recurrent disease. In our four patients, the intraoperative findings were concordant with the endosonographic findings both in terms of localization and size of the lesions. However, it should be stressed that US imaging is not specific and that histology is needed as confirmation [19]. In the treatment of endometriosis, the patient’s age should be considered. In the perimenopausal period, endometriosis has a tendency to regress. As consequence, surgical excision can be limited to the lesion, preserving
Tech Coloproctol
Fig. 3 a 3D endoanal ultrasound reconstruction on the coronal plane shows the endometriosic lesion (L) invading the distal EAS (IAS internal anal sphincter). b Excised endometriosis focus
the anal sphincters, and hormonal therapy is not administered [20]. For patients of reproductive age, published studies recommend wide excision of anal and perianal endometriosis, followed by hormonal therapy [5]. We adopted this management in our cases, and no recurrences were reported at follow-up.
Conclusions EAUS and TPUS are useful imaging modalities for assessing anal and perianal endometriosis and excluding more common causes as perianal abscess and anal cancer. US helps to plan adequate management and can be used in follow-up after treatment. Conflict of interest
None.
Fig. 4 a, b Transperineal ultrasound showing an hypoechoic, vascularized area in the anterior and left lateral side of the anal canal, with posterior wall enhancement. c Endoanal ultrasound confirms the presence of the lesion (L) in the left lateral ischiorectal space, adjacent to the EAS (IAS internal anal sphincter)
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References 1. Allaire C (2006) Endometriosis and infertility: a review. J Reprod Med 51:164–168 2. Treloar SA, Wicks J, Nyholt DR et al (2005) Genome wide linkage study in 1.176 affected sister pair families identifies a significant susceptibility locus for endometriosis on chromosome 10q26. Am J Hum Genet 3:365–376 3. Benagiano G, Brosens I (1991) The history of endometriosis: identifying the disease. Hum Reprod 6:963–968 4. Riedel HH, Semm K (1982) Extragenital endometriosis and sterility-clinical experiences with a 3-step-therapy. Prog Clin Biol Res 112:187–194 5. Kanellos I, Kelpis T, Zaraboukas T, Betsis D (2001) Perineal endometriosis in episiotomy scar with anal sphincter involvement. Tech Coloproctol 5:107–108 6. Sayfan J, Benosh L, Segal M, Orda R (1991) Endometriosis in episiotomy scar with anal sphincter involvement. Report of a case. Dis Colon Rectum 34:713–716 7. Odobasic A, Pasic A, Iljazovic-Latifagic E et al (2010) Perineal endometriosis: a case report and review of the literature. Tech Coloproctol 14:25–27 8. Barisic GI, Krivokapic ZV, Jovanovic DR (2006) Perineal endometriosis in episiotomy scar with anal sphincter involvement: report of two cases and review of the literature. Int Urogynecol J Pelvic Floor Dysfunct 17:646–649 9. Santoro GA, Fortling B (2007) The advantages of volume rendering in three-dimensional endosonography of the anorectum. Dis Colon Rectum 50:359–368 10. Sudoł-Szopin´ska I, Kołodziejczak M, Szopin´ski TR (2011) The accuracy of a postprocessing technique—volume render mode— in three-dimensional endoanal ultrasonography of anal abscesses and fistulas. Dis Colon Rectum 54:238–244 11. Roos AM, Abdool Z, Sultan AH, Thakar R (2011) The diagnostic accuracy of endovaginal and transperineal ultrasound for
123
12.
13.
14.
15.
16.
17.
18.
19.
20.
detecting anal sphincter defects: the PREDICT study. Clin Radiol 66:597–604 Kołodziejczak M, Sudoł-Szopin´ska I, Wilczyn´ska A, Bierca J (2012) Utility of transperineal and anal ultrasonography in the diagnostics of hidradenitis suppurativa and its differentiation from a rectal fistula. Postepy Hig Med Dosw 14:838–842 Coutinho A, Bittencourt LK, Pires CE et al (2011) MR imaging in deep pelvic endometriosis: a pictorial essay. Radiographics 31:549–567 Gui B, Valentini AL, Ninivaggi V, Marino M, Iacobucci M, Bonomo L (2014) Deep pelvic endometriosis: don’t forget round ligaments. Review of anatomy, clinical characteristics and MR imaging features. Abdom Imaging 39:622–632 Bazot M, Darai E, Hourani R et al (2004) Deep pelvic endometriosis: MR imaging for diagnosis and prediction of extension of disease. Radiology 232:379–389 Bliss DZ, Mellgren A, Whitehead WE et al (2013) Assessment and conservative management of faecal incontinence and quality of life in adults. In: Abrams P, Cardozo L, Khoury S, Wein A (eds) 5th international consultation on incontinence. Paris: ICUDEAU 16:1443-1486 Gonza´lez-Longoria G, Mejı´a-Ovalle RR, Salinas-Arago´n E, Jime´nez-Gonza´lez A, Gonza´lez-Contreras HQ (2011) Perineal endometriosis with anal external sphincter involvement: a casereport. Rev Gastroenterol Mex 76:173–177 Del Frate C, Girometti R, Pittino M, Del Frate G, Bazzocchi M, Zuiani C (2006) Deep retroperitoneal pelvic endometriosis: MR imaging appearance with laparoscopic correlation. Radiographics 26:1705–1718 Pishvaian AC, Ahlawat SK, Garvin D, Haddad NG (2006) Role of EUS and EUS-guided FNA in the diagnosis of symptomatic rectosigmoid endometriosis. Gastrointest Endosc 63:331–335 Leyland N, Casper R, Laberge P, Singh S (2010) Endometriosis: diagnosis and management. Soc Obstet Gynaecol Can 32(Suppl 2):1–33
TECHNICAL NOTE
Ultrasonographic evaluation of anal endometriosis: report of four cases M. Kołodziejczak • I. Sudoł-Szopin´ska • G. A. Santoro • K. Bielecki • A. Wia˛czek
Received: 1 January 2014 / Accepted: 30 May 2014 Ó Springer-Verlag Italia Srl 2014
Abstract Background The presence of endometriosis in the anal canal and perianal tissues is rare and difficult to suspect at clinical examination. We report our experience with preoperative ultrasound evaluation of four cases of anal endometriosis. Methods Four patients were evaluated by transperineal and high-resolution three-dimensional endoanal ultrasonography. Results In 3 of 4 women, the lesions involved old episiotomy scars. Anal endometriosis appeared as hypoechoic cystic lesions with areas of microcalcification, not well delimited and highly vascularized. The lesions either involved the anal sphincter (n = 2, one within the rectovaginal septum) or were localized superficially in the ischiorectal space (n = 2). Surgery and pathologic exam confirmed the ultrasonographic findings. Conclusions Ultrasonographic findings of anal endometriosis are characteristics and may allow accurate preoperative staging of the disease.
M. Kołodziejczak K. Bielecki A. Wia˛czek Department of General Surgery, Sub-Department of Proctology, Hospital at Solec, 00-382 Warsaw, Poland I. Sudoł-Szopin´ska Department of Radiology, Institute of Rheumatology, Warsaw, Poland I. Sudoł-Szopin´ska Department of Diagnostic Imaging, Warsaw Medical University, Spartan´ska 1 St. 8, 02-637 Warsaw, Poland G. A. Santoro (&) Pelvic Floor Unit, I Department of Surgery, Regional Hospital, Piazzale Ospedale 1, 31100 Treviso, Italy e-mail: [email protected]
Keywords Anal sphincters Endoanal ultrasonography Endometriosis Transperineal ultrasonography
Introduction Endometriosis is a chronic disease, affecting 10–15 % of women of reproductive age, which characterized by the ectopic localization of the hormonally active uterine endometrium [1]. It has an unclear and complex pathophysiology, including different risk factors: history of gynecological surgery, high estrogen levels, early menarche, short menstrual cycles (\27 days), menstrual bleeding for [7 days, and immune system disorders [2]. According to the implantation theory, the endometrial cells travel through the fallopian tubes or may also reach remote organs via blood and lymphatic vessels [3]. Typical localizations of endometriosis are the uterosacral ligaments and ovaries (approximately 60 %), the pouch of Douglas (25 %), the vesicouterine space (in the form of tarry cysts) (20 %), the round ligament of the uterus (11 %), and the bowel (7 %) [4]. Rarely, endometriotic foci can be found in the anal canal and perianal tissues [5–8]. Symptoms of endometriosis are pain in the lower pelvis or in the perineum (particularly during the menstrual cycle), menstrual disorders, pain during sexual intercourse, and infertility. Preoperative imaging of anal endometriosis in order to determine whether the foci involve the anal sphincters or are limited to the perianal space is fundamental for planning adequate surgical treatment. In this paper, we report our experience with four cases assessed by transperineal (TPUS) and high-resolution three-dimensional (3D) endoanal ultrasound (EAUS).
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Imaging techniques Endoanal ultrasonography (EAUS) EAUS was performed with the patient placed in the left lateral decubitus, using a 360° rotating, mechanical, multifrequencies (frequency range 6–16 MHz) transducer with automatic, computed-controlled (300 transaxial images, over a distance of 60 mm in 60 s), high-resolution 3D acquisition (type 2050, BK Medical Analogic Corporation, Herlev, Denmark) [9, 10]. In the axial plane, the anal canal was divided into three levels of assessment referring to the following anatomic structures: (1) upper level: the hyperechoic sling of the puborectalis muscle (PR) and the complete hypoechoic ring of the internal anal sphincter (IAS); (2) middle level: the superficial, mixed echogenicity ring of the external anal sphincter (EAS), the conjoined longitudinal layer, the IAS, and the transverse superficial perinei muscles; and (3) lower level: the subcutaneous EAS. The 3D reconstruction was useful in assessing the anatomic characteristics of the anal canal in the longitudinal and coronal planes (MPR = multiplanar reconstructions).
proctological disease were reported. Her obstetrical history was characterized by a vaginal delivery 11 years before. On rectal examination, a mobile and painful nodule of approximately 4 cm was found in the posterior, right lateral quadrant of the anal canal, at 1 cm from the anal verge. The resting and squeeze pressures of the sphincter muscles were normal. Proctoscopy revealed a normal mucosal surface. On TPUS, a subcutaneous, hypoechoic, and highly vascularized lesion was revealed. It was not well circumscribed, and its dimensions were 2.1 9 3.5 cm (Fig. 1a). On EUAS, an hypoechoic lesion was visualized in the perianal tissue, adjacent to but noninvading the outer border of the subcutaneous EAS (Fig. 1b). Based on the ultrasound (US) findings that confirmed the absence of a chronic abscess, the patient underwent surgery. A hard mass of 3 9 5 cm, localized in the right perianal space, adjacent to the anal sphincter, was excised. Histological analysis of the specimen revealed a focus of endometriosis. The patient was given hormonal therapy for 10 months after surgery. At the clinical and US follow-up at 2 years, no recurrence was found. Case 2
We report four cases of anal endometriosis assessed with both 3D-EAUS and TPUS (Table 1). The protocol was approved by the Medical University’s review board, and all participants gave written informed consent.
A 42-year-old woman presented with an anovaginal fistula. She had had two vaginal deliveries with episiotomy. At clinical assessment, the vaginal orifice of the fistula was located at 1.5 cm from the vaginal verge and the anal orifice at 2 cm from the anal margin. Proctoscopy (up to 20 cm) confirmed the presence of the anal opening. The surface of the rectal mucosa was normal. On EAUS, both the anovaginal tract and the internal opening (IO) at the level of the anorectum were visualized. No collection was demonstrated (Fig. 2). At surgery, the fistula with its anal opening was excised and the defect was closed with an anocutaneous advancement flap. The vaginal opening was also sutured with a series of absorbable stitches. There were no postoperative complications. Histological analysis revealed the presence of fibrous tissue and chronic inflammation. Foci of endometriosis were found in the stroma. The patient, following gynecological consultation, received hormonal therapy. At 3-year follow-up, clinical examination and ultrasonographic assessment confirmed the healing of the fistula and the presence of a scar.
Case 1
Case 3
A 34-year-old woman was admitted to our department with suspected anal abscess. She complained of a painful, perianal mass, without purulent discharge or rectal bleeding, first noticed 12 months prior, ineffectively treated with antibiotics. No fever, trauma of the perineum, or any other
A 33-year-old woman with a 2 cm painful nodule localized in the episiotomy scar from her vaginal delivery 12 years prior was admitted to our department for surgical treatment. She did not complain of other symptoms. On TPUS, an hypoechoic, not well delimited, vascularized area
Transperineal ultrasonography (TPUS) Transperineal ultrasound was performed with the patient placed in dorsal lithotomy, with the hips flexed and abducted, using a 2D linear array, multifrequencies (frequency range 5–12 MHz) transducer (type 8811, BK Medical Analogic Corporation). The probe was positioned in the perineum, slightly anterior to the anus, and was rotated in order to provide sagittal, coronal, and oblique sectional imaging of the anal canal and perianal tissues. Power Doppler (PWD) option allowed evaluation of vascularity of the lesions [11, 12].
Case reports
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Tech Coloproctol Table 1 Clinical and ultrasonographic findings in patients with anal and perianal endometriosis Patient no.
History
Clinical examination
Transperineal ultrasonography
3D endoanal ultrasonography
Surgical treatment
1.
34-year-old woman with suspected anal abscess
Mobile and painful 4 cm nodule, in the posterior, right lateral quadrant of the anal canal, at 1 cm from the anal margin
Subcutaneous, 2.1 9 3.5 cm size, hypoechoic and highly vascularized lesion, not well circumscribed
Hypoechoic lesion in the perianal tissue, adjacent but noninvading the outer border of the subcutaneous EAS
Excision of a firm lesion, 3 9 5 cm, located in the right perianal space, adjacent to the anal sphincter
2.
42-year-old woman with an anovaginal fistula
Anovaginal fistula at 1.5 cm from the vaginal verge and at 2 cm from the anal margin
Subcutaneous fistulous tract in the perineal tissue
Anovaginal tract with visualization of the internal anal opening. No other collection. Regular EAS
Excision of the fistula with its internal anal opening; anocutaneous advancement flap; suture of the vaginal opening
3.
33-year-old woman with a 2 cm painful nodule located in the episiotomy scar
2 cm painful nodule in the episiotomy scar
Hypoechoic, not well delimited, vascularized area. 2 9 3 cm, with small cysts inside, located the anterior, right lateral quadrant of the anal canal
Hypoechoic lesion involving the anterior 1/3 of the superficial EAS, the adjacent intersphincteric space and the perineal tissue
Excision of a nodule, in the deeper part of the rectovaginal septum, with a small portion of the anterior EAS. Repair of the EAS with ‘‘end-toend’’ technique
4.
39-year-old woman with recurrence of endometriosis located in the episiotomy scar
4 cm nodule in the anterior and left lateral quadrants of the anal canal
Two hypoechoic nodules, 8 and 12 mm, not well delimited, with posterior wall enhancement, highly vascularized, with small cystic areas inside and microcalcifications
Hypoechoic lesion 4 9 2 cm in the anterior and left lateral quadrants of the anal canal, involving the ischiorectal fossa, the perineum and the lateral border of the EAS
Excision of two nodules, dissected from the fibers of the EAS
EAS external anal sphincter
2 9 3 cm with small cysts was detected in the anterior, right lateral quadrant of the anal canal. On 3D EAUS, the lesion involved the anterior 1/3 of the superficial EAS, the adjacent intersphincteric space, and the perineal tissue. No fistulous tracts were visualized (Fig. 3a). At surgery, the nodule, which penetrated into the deeper part of the rectovaginal septum, was completely excised with a small portion of the anterior EAS (Fig. 3b). There was no invasion of the rectal and vaginal walls. The anal sphincter was repaired with an ‘‘end-to-end’’ technique. The patient was discharged on the sixth postoperative day. Histological analysis demonstrated an endometriotic lesion. The patient received hormonal treatment. At 12-month follow-up, no recurrence was revealed. Case 4 A 39-year- old woman underwent excision of 2.5 cm focus of endometriosis in her episiotomy scar, 12 years after a vaginal delivery with episiotomy. At 6-month follow-up, she presented a 1 cm recurrence that was treated with hormonal therapy. However, at 1-year follow-up, the lesion had grown to approximately 4 cm.
On proctologic examination, a nodule of 4 cm was found in the anterior and left lateral quadrants of the anal sphincters. Proctoscopy up to 20 cm was normal. Transperineal US demonstrated two nodules, 8 and 12 mm in diameter. The lesions were hypoechoic with posterior wall enhancement, not well delimited, highly vascularized, with microcalcifications and small cystic areas (Fig. 4a, b). At EAUS, the lesion measured 4 9 2 cm and was localized in the anterior and left lateral quadrants of the anal canal. It involved the ischiorectal fossa, the perineum, and the lateral border of the subcutaneous and superficial parts of the EAS (Fig. 4c). At surgery, these two nodules were dissected from the fibers of the EAS and were completely excised. Histological analysis confirmed the recurrence of endometriotic foci. The patient was discharged on postoperative day 2.
Discussion The presence of endometriosis in the anal canal, in the ischiorectal fossa, and perianal tissues is rare and difficult to suspect at clinical examination. Detailed imaging is
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Fig. 2 Endoanal ultrasound showing the internal orifice (IO) of the anovaginal fistula in the anterior part of the middle level of the anal canal (EAS external anal sphincter)
Fig. 1 a Transperineal ultrasound shows an hypoechoic, highly vascularized, and not well-circumscribed lesion, 21 9 35 mm, at the right side of the anus. b At endoanal ultrasound, the lesion (L) appears in contact, but not invading, the subcutaneous part of the EAS
mandatory in order to exclude more common causes as perianal abscesses or anal tumors and for planning adequate management, and defining the size and position of the lesion as well as its relationship with the anal sphincter muscles. Magnetic resonance imaging (MRI) provides important information on the extension of endometriotic disease, helping to identify deep pelvic endometriosis in the retrocervical region, uterosacral and round ligaments, rectosigmoid, vagina, urinary tract, and other subperitoneal and extraperitoneal pelvic sites [13, 14]. Bazot et al. [15] reported that the sensitivity, specificity, positive and negative predictive values, and accuracy of MRI for deep pelvic endometriosis were 90.3, 91, 92, 89, and 90.6 %, respectively. However, EAUS has been defined the gold standard for assessment of the anal canal [16, 17]. 3D US with high spatial resolution allows multiplanar evaluation
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and good tissue characterization of the sphincter muscles, the perianal tissues, and the rectovaginal septum [9]. In addition, TPUS allows the assessment of the vascularity by using PWD. MRI and US should not be considered as alternative modalities, but can be combined in selected cases of deep infiltrating endometriosis, thereby facilitating accurate diagnosis and adequate treatment [18]. In our study, at TPUS, anal endometriosis was most frequently visualized as an hypoechoic or mixed echogenicity lesion, with areas of microcalcification or small cysts, not well delimited and highly vascularized. The relationship of the lesion with the anal sphincters and the rectal wall could be evaluated by EAUS. In Case 3, the lesion involved the EAS and these data were confirmed intraoperatively. The focus was excised, and the muscle was repaired. In Case 4, the lesion was in adjacent to but not invading the outer border of the EAS. At surgery, we could dissect the focus from the EAS, without removing muscle fibers. EAUS also helps in identifying anal sepsis or a fistula. In Case 2, an anovaginal fistula was demonstrated and subsequently treated by an anocutaneous advancement flap. Moreover, US is important in follow-up to confirm presence of scar tissue or recurrent disease. In our four patients, the intraoperative findings were concordant with the endosonographic findings both in terms of localization and size of the lesions. However, it should be stressed that US imaging is not specific and that histology is needed as confirmation [19]. In the treatment of endometriosis, the patient’s age should be considered. In the perimenopausal period, endometriosis has a tendency to regress. As consequence, surgical excision can be limited to the lesion, preserving
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Fig. 3 a 3D endoanal ultrasound reconstruction on the coronal plane shows the endometriosic lesion (L) invading the distal EAS (IAS internal anal sphincter). b Excised endometriosis focus
the anal sphincters, and hormonal therapy is not administered [20]. For patients of reproductive age, published studies recommend wide excision of anal and perianal endometriosis, followed by hormonal therapy [5]. We adopted this management in our cases, and no recurrences were reported at follow-up.
Conclusions EAUS and TPUS are useful imaging modalities for assessing anal and perianal endometriosis and excluding more common causes as perianal abscess and anal cancer. US helps to plan adequate management and can be used in follow-up after treatment. Conflict of interest
None.
Fig. 4 a, b Transperineal ultrasound showing an hypoechoic, vascularized area in the anterior and left lateral side of the anal canal, with posterior wall enhancement. c Endoanal ultrasound confirms the presence of the lesion (L) in the left lateral ischiorectal space, adjacent to the EAS (IAS internal anal sphincter)
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References 1. Allaire C (2006) Endometriosis and infertility: a review. J Reprod Med 51:164–168 2. Treloar SA, Wicks J, Nyholt DR et al (2005) Genome wide linkage study in 1.176 affected sister pair families identifies a significant susceptibility locus for endometriosis on chromosome 10q26. Am J Hum Genet 3:365–376 3. Benagiano G, Brosens I (1991) The history of endometriosis: identifying the disease. Hum Reprod 6:963–968 4. Riedel HH, Semm K (1982) Extragenital endometriosis and sterility-clinical experiences with a 3-step-therapy. Prog Clin Biol Res 112:187–194 5. Kanellos I, Kelpis T, Zaraboukas T, Betsis D (2001) Perineal endometriosis in episiotomy scar with anal sphincter involvement. Tech Coloproctol 5:107–108 6. Sayfan J, Benosh L, Segal M, Orda R (1991) Endometriosis in episiotomy scar with anal sphincter involvement. Report of a case. Dis Colon Rectum 34:713–716 7. Odobasic A, Pasic A, Iljazovic-Latifagic E et al (2010) Perineal endometriosis: a case report and review of the literature. Tech Coloproctol 14:25–27 8. Barisic GI, Krivokapic ZV, Jovanovic DR (2006) Perineal endometriosis in episiotomy scar with anal sphincter involvement: report of two cases and review of the literature. Int Urogynecol J Pelvic Floor Dysfunct 17:646–649 9. Santoro GA, Fortling B (2007) The advantages of volume rendering in three-dimensional endosonography of the anorectum. Dis Colon Rectum 50:359–368 10. Sudoł-Szopin´ska I, Kołodziejczak M, Szopin´ski TR (2011) The accuracy of a postprocessing technique—volume render mode— in three-dimensional endoanal ultrasonography of anal abscesses and fistulas. Dis Colon Rectum 54:238–244 11. Roos AM, Abdool Z, Sultan AH, Thakar R (2011) The diagnostic accuracy of endovaginal and transperineal ultrasound for
123
12.
13.
14.
15.
16.
17.
18.
19.
20.
detecting anal sphincter defects: the PREDICT study. Clin Radiol 66:597–604 Kołodziejczak M, Sudoł-Szopin´ska I, Wilczyn´ska A, Bierca J (2012) Utility of transperineal and anal ultrasonography in the diagnostics of hidradenitis suppurativa and its differentiation from a rectal fistula. Postepy Hig Med Dosw 14:838–842 Coutinho A, Bittencourt LK, Pires CE et al (2011) MR imaging in deep pelvic endometriosis: a pictorial essay. Radiographics 31:549–567 Gui B, Valentini AL, Ninivaggi V, Marino M, Iacobucci M, Bonomo L (2014) Deep pelvic endometriosis: don’t forget round ligaments. Review of anatomy, clinical characteristics and MR imaging features. Abdom Imaging 39:622–632 Bazot M, Darai E, Hourani R et al (2004) Deep pelvic endometriosis: MR imaging for diagnosis and prediction of extension of disease. Radiology 232:379–389 Bliss DZ, Mellgren A, Whitehead WE et al (2013) Assessment and conservative management of faecal incontinence and quality of life in adults. In: Abrams P, Cardozo L, Khoury S, Wein A (eds) 5th international consultation on incontinence. Paris: ICUDEAU 16:1443-1486 Gonza´lez-Longoria G, Mejı´a-Ovalle RR, Salinas-Arago´n E, Jime´nez-Gonza´lez A, Gonza´lez-Contreras HQ (2011) Perineal endometriosis with anal external sphincter involvement: a casereport. Rev Gastroenterol Mex 76:173–177 Del Frate C, Girometti R, Pittino M, Del Frate G, Bazzocchi M, Zuiani C (2006) Deep retroperitoneal pelvic endometriosis: MR imaging appearance with laparoscopic correlation. Radiographics 26:1705–1718 Pishvaian AC, Ahlawat SK, Garvin D, Haddad NG (2006) Role of EUS and EUS-guided FNA in the diagnosis of symptomatic rectosigmoid endometriosis. Gastrointest Endosc 63:331–335 Leyland N, Casper R, Laberge P, Singh S (2010) Endometriosis: diagnosis and management. Soc Obstet Gynaecol Can 32(Suppl 2):1–33
