Tech Coloproctol DOI 10.1007/s10151-014-1146-8
ORIGINAL ARTICLE
A laparoscopic approach to benign retrorectal tumors J. L. Zhou • B. Wu • Y. Xiao • G. L. Lin W. Z. Wang • G. N. Zhang • H. Z. Qiu
•
Received: 5 January 2014 / Accepted: 25 March 2014 Ó Springer-Verlag Italia 2014
Abstract Background Retrorectal tumors (RTs) are rare in adults. Their surgical excision is often difficult because of their anatomic location. The aim of this study was to evaluate the results of straight laparoscopic resection of RTs in our institution. Methods Eight patients (six women and two men) with benign RTs were treated by laparoscopic resection in our tertiary care center between September 2012 and June 2013. Exclusion criteria included malignant tumors, lesions with fistula formation, and anterior sacral meningoceles. Clinical data, imaging features, operative details, pathological results, and treatment outcomes were reviewed and analyzed. Results Eight cases of benign RT with an average diameter of 8.9 ± 1.7 cm were treated by a straight laparoscopic procedure. The mean operative time was 122 ± 36 min, and the average intraoperative blood loss was 46 ± 33 ml. The median postoperative stay was 5 days (range 3–8 days), and all patients were discharged without serious complications. During a median follow-up of 11 months, no tumor recurrence was observed.
J. L. Zhou and B. Wu have contributed equally to this study. J. L. Zhou B. Wu Y. Xiao G. L. Lin G. N. Zhang H. Z. Qiu (&) Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Dong Dan, Dong Cheng District, Beijing 100730, China e-mail: [email protected] W. Z. Wang Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Beijing, China
Conclusions In our experience, a laparoscopic approach is safe for removing benign tumors in the retrorectal space. This approach may provide access to this difficult-to-reach space and has the advantages of allowing excellent visualization, meticulous dissection, less morbidities, and fast recuperation. Keywords Benign
Laparoscopy Adults Retrorectal tumor
Introduction Retrorectal tumors (RTs), a group of heterogeneous and uncommon lesions, develop in the retrorectal space. This potential space, which lies anterior to the sacrum and coccyx and posterior to the rectum, is also called the presacral space [1, 2]. RTs are rare in adults, with an estimated incidence of one in 40,000–63,000 admissions to large referral centers [1, 3, 4]. Benign RTs may have potential for malignant degeneration [5, 6]. After enlarging, they may interfere with the pelvic outlet, causing difficult or incomplete evacuation of stools, and can even lead to obstructive dystocia in women of childbearing age [2, 6, 7]. Developmental cysts are associated with a risk of secondary infection and may form fistulas to the sacrococcygeal skin or into the rectum [6, 8]. It is commonly believed that RTs should be surgically removed once diagnosed [1, 5, 9, 10]. Their anatomic location makes RTs hard to reach and so they have traditionally been accessed through transsacral, transabdominal, or a combined abdominosacral route. Each approach has advantages and disadvantages. A laparoscopic approach to RTs was first reported in the 1990s. Recently, successful laparoscopic resections of
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No 3 None 20 F 8
BMI body mass index, DRE digital rectal examination, M male, F female, CT computed tomography, MRI magnetic resonance imaging, US ultrasound
80 US ? CT 9.4 9 4.1 9 3.1 ? None Laparoscopic oophorocystectomy None Leiomyoma 20
F 7
43
No 5 None 40 115 US ? MRI 9.3 9 9.0 9 7.2 ? None None None Epidermoid cyst 19.2
F 6
27
No 5 None 50 150 US ? CT 9.0 9 9.0 9 8.0 ? None None None Mature teratoma 22.5
M 5
15
No 7 None 30 150 CT 10.0 9 5.4 9 4.5 ? Hypertension, hyperlipoidemia, and Cushing’s syndrome Laparotomy for mature teratoma of bladder None Mature teratoma 20.8
F 4
23
No 5 None 30 85 US ? CT 7.0 9 6.6 9 4.6 ? None Cesarean section None Epidermoid cyst 21.2
F 3
29
No 3 None 20 180 MRI 11.8 9 8.8 9 8.0 ? None Open hysteromyomectomy None Epidermoid cyst 21.8
F 2
47
No 4 None 120 95 US ? CT 6.3 9 6.2 9 5.8 Hypertension
M
63
26
Schwannoma
Lower abdominal pain
Laparoscopic exploration
–
No 5 None 60 120 CT 8.0 9 7.5 9 7.1 ? Hypertension and coronary heart disease None None Schwannoma 23.9
Operating time (min) Diagnosis Size (cm) DRE Comorbidities Previous abdominal surgery Symptom Pathologic diagnosis
1
All operations were performed by two colorectal surgeons with skill in laparoscopic colorectal surgeries. The patient was placed in a steep Trendelenburg position. Five ports (two 10-mm and three 5-mm ports) placed in a similar pattern as that for laparoscopic rectal cancer resection were used. In most cases, dissection began from the right side of
BMI
Surgical technique
Age (years)
Results are presented as median values or mean ± standard deviation (SD) for continuous variables. The independentsamples t test was used to assess tumor size differences between this and two other series. All tests for statistical significance were two-tailed, the level of significance being set at p \ 0.05. All data were analyzed using SPSS software (version 19.0 for Windows, SPSS, Chicago, IL, USA).
Sex
Statistical analysis
No.
In the period from September 2012 to June 2013, eight patients with benign RTs underwent straight laparoscopic resection performed by two experienced colorectal surgeons at our tertiary care center. The preoperative diagnoses were made on the basis of clinical features and imaging including ultrasound (US), computerized tomography (CT) scan, or magnetic resonance imaging (MRI). During the study period, laparoscopic approaches were used for retrorectal lesions identified as benign regardless of tumor size or extent of the lesion. Lesions identified as malignant tumors, benign lesions with severe infection or fistula formation, and anterior sacral meningoceles (ASM) were excluded. Data including the patients’ sex, age, clinical presentation, physical examination, imaging characteristics, final pathological diagnosis, operative details, intra- and postoperative complications, postoperative length of stay, mortality, and recurrence rates were recorded and summarized (Table 1).
Table 1 Description of retrorectal tumor cases treated by the laparoscopic excision including patient demographics and clinical variables
Materials and methods
Intraoperative blood loss (ml)
Complications
Postoperative length of stay (days)
benign RTs have been reported, mostly as initial attempts [11–27]. In this study, we reviewed eight cases of benign RT treated with laparoscopic surgery. We examined patient characteristics, operative details, perioperative morbidities, and outcomes to assess the feasibility, safety, and oncological outcome (in terms of incomplete excision or tumor recurrence) of the laparoscopic approach. Selected aspects of the diagnosis of RTs and surgical procedures are also discussed.
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Recurrence
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the upper mesorectum above the sacral promontory. After the right retroperitoneum had been opened with a harmonic scalpel, the flimsy areolar tissue in the plane between the visceral mesorectal fascia and the parietal pelvic fascia was identified. Dissection proceeded medially and downward along this plane to mobilize the rectum from the retrorectal space. Care was taken to identify and safeguard the ureters and pelvic autonomic nerves. Left-sided tumors were accessed from the left. For large lesions, opening the retroperitoneum on both sides provided better exposure. With the rectum retracted upward and anteriorly, dissection continued downward until the lesion was revealed (Fig. 1a). The tumor was carefully divided from the pelvic side wall and adjacent structures along its capsule (Fig. 1b). It was sometimes difficult to distinguish the borders of secondarily infected retrorectal cysts. The correct plane was carefully identified to prevent cutting into the mesorectum and causing bleeding or injuring the rectal wall. When a cystic lesion occupied too much of the working space, it was partially collapsed by aspiration to allow dissection to continue. The collapsed cyst was moved aside to reveal its remaining attachments, greatly
facilitating further dissection (Fig. 1c). Retrorectal developmental cysts usually have a dense attachment to the coccyx and anterior surface of the lower sacrum (Fig. 2) and may extend into or below the pelvic floor muscles. In such cases, dissection was continued downward close to the tumor capsule. Attachment to the levator ani muscles was severed, and the inferior end of the lesion dissected off until the entire lesion could be removed (Fig. 1c, d), taking care to distinguish the muscle fibers of the levator ani from those of the rectal wall so as not to injure the latter. The specimen was extracted using a laparoscopic retrieval bag (Endocatch Gold 10 mm; Surgical Corporation, Norwalk, CT, USA) via a prolonged trocar incision and the retrorectal space adequately irrigated. A drain was placed in the surgical anatomic space and brought out through a trocar incision. Before surgery, all patients were fully informed about the procedure and potential alternatives, risks, and complications, and provided written informed consent. Bowel preparation with polyethylene glycol electrolytes powder was performed the afternoon before the operation and a single dose of prophylactic antibiotic given just prior
Fig. 1 Details of laparoscopic resection of a retrorectal epidermoid cyst (case 7). a The top of the retrorectal cyst (arrow 1) was revealed from behind the mesorectum (arrow 2) by harmonic dissection between the posterior mesorectal fascia and the presacral fascia. b Care was given to dissect the cyst (arrow 1) along its capsule to prevent injury to the rectal wall (arrow 2). c Dissection continued
downward between the collapsed cyst (encircled by the curved line) and the musculature of the pelvic floor (white arrows) to enter the ischiorectal space (black arrow), to reach the lower end of the cyst. d Finally, the collapsed cyst (black arrow) was cut from its attachment to the levator ani muscle (white arrow)
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Fig. 2 Sagittal T2-weighted MR image showing the retrorectal epidermoid cyst in case 7, which is adherent to the coccyx and lower sacrum and extends below the levator ani muscle
to induction of anesthesia. Postoperative interventions included intravenous fluid and on-demand analgesia. Early mobilization was encouraged. Oral water intake started the day after surgery, and a liquid diet was initiated once the patient passed flatus or had a bowel movement. Patients were discharged according to the hospital’s discharge criteria. Follow-up Follow-up was conducted by interview in the outpatient clinic and by telephone. All patients were required to return to the outpatient clinic 1 and 6 months postoperatively, to attend for long-term follow-up thereafter, and to return to our hospital whenever there was any suspicion of disease recurrence. Digital rectal examination (DRE) and pelvic US were performed routinely to detect recurrence. The long-term complications and rate of recurrence were recorded at follow-up.
Results The series of eight patients consisted of six women and two men with a median age of 36 years (range 15–75 years). The diagnoses on final pathological examinations were three epidermoid cysts (including one infected cyst), two mature teratomas, two schwannomas, and one leiomyoma; all were benign (Table 1).
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Seven patients were asymptomatic, their lesions having been detected incidentally during imaging studies performed for other purposes. One 63-year-old woman with a schwannoma (patient 2) had noticed lower abdominal pain and fullness 1 month before discovery of the tumor. On physical examination, a retrorectal mass (behind or beside the rectum) was detected by DRE in seven patients. The mean distance, measured by DRE, from the lower end of the mass to the anal verge was 4.1 ± 1.7 cm. In six patients, the upper end of the lesion could not be reached on DRE. In one patient with schwannoma, the tumor could not be reached on DRE but it was detectable as an immobile round hard pelvic mass by abdominal palpation. Pelvic US was performed in five patients. Six patients underwent spiral CT scan and another two underwent MRI, which clearly defined the location of the tumors and their relationships with adjacent structures. All tumors were shown to be well-defined retrorectal soft-tissue masses that did not invade of adjacent organs. According to the imaging studies, the largest tumors ranged from 6.3 to 11.8 cm in diameter (mean 8.9 ± 1.7 cm). Straight laparoscopy was completed in all patients, neither conversion to open surgery nor combination with other approaches being required. All five developmental cysts (three epidermoid cysts and two mature teratomas) were decompressed during surgery to improve exposure and facilitate dissection, either by intentionally puncturing them followed by aspiration with a suction device or after accidental rupture during dissection. Any semisolid material spilled from the cysts was removed by suction and repeated lavage to prevent postoperative complications such as peritonitis, adhesion formation, and wound complications. Two of the epidermoid cysts (patients 3 and 7) extended beneath the pelvic floor muscles, necessitating the continuation of dissection downward between the cystic capsule and levator ani muscles to enter the ischiorectal space. Macroscopically complete tumor removal was achieved in all eight cases. No injury to the rectal wall or other pelvic viscera occurred. The mean operating time was 122 ± 36 min, and intraoperative blood loss was insignificant (mean 46 ± 33 ml). Median postoperative length of stay was 5 days (range 3–8 days). Postoperatively, urinary tract infection occurred on day 5 in one patient (patient 5) with the comorbidity of ectopic adrenocorticotropic hormone syndrome. Antibiotics were administered with relief of symptoms within 2 days. In the other seven patients, postoperative fever of \38.5 °C was observed only within 48 h of surgery. No bowel damage, postoperative bowel obstruction, surgical site infection, or wound complication occurred; there was no perioperative mortality. During a median follow-up of 11 months (range 4–14 months), no residual lesion or tumor recurrence was
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observed. Two patients complained that they felt no urge to defecate and had constipation; these symptoms improved with laxatives and increased dietary fiber. No other longterm complication was observed.
Discussion The retrorectal space is an area of development of multiple embryologic structures; a heterogeneous group of benign and malignant tumors can arise from their remnants [5, 28]. Most RTs are benign, and these benign tumors are most often developmental cysts (epidermoid, dermoid and tailgut cysts, and benign teratomas) and schwannomas. According to the previous reports, RTs are rare [1, 3]. However, because many remain clinically silent, the true prevalence of these lesions is unknown [4]. This study included eight patients with benign RTs treated at our institution within a period of 10 months. Seven were asymptomatic, their lesions being detected accidentally during imaging for other reasons. With the increasing performance of imaging studies during health checks, more asymptomatic cases will likely be uncovered. Symptoms of benign RTs are usually nonspecific or related to local mass effect [8]. Patients may complain of pain, a sensation of downward pressure in the pelvis or rectum, or a change in bowel habits [29]. Patients with large lesions may present with difficult or incomplete defecation or even dysuria. Among women of childbearing age, such masses occasionally obstruct labor, necessitating Cesarean section [2, 5]. It is generally considered that RTs should be surgically removed once diagnosed, even when asymptomatic and benign [1, 5, 9, 10]. Almost all RTs can be palpated on DRE [1], which can determine their size, location, consistency, and relationship with the rectal wall and surrounding structures. DRE is therefore the simplest, most efficient method for identifying these lesions. However, because of their soft texture and the smoothness of the overlying rectal mucosa, they can escape detection on DRE. Thus, careful palpation by sweeping the examining finger across the mucosal surface is necessary. Retrorectal tumors are readily revealed by transabdominal US. However, inexperienced sonographers can misdiagnose retrorectal cysts as adnexal lesions, especially when the ovaries are not visualized precisely. In this series, case 2 was initially misdiagnosed as having a left ovarian tumor and underwent an unfruitful laparoscopic exploration in the gynecology department. Similar cases have been reported by other authors [10, 30]. Spiral CT scan and MRI, especially sagittal plane images (Fig. 2), are very helpful in defining RTs. They demonstrate these tumors’ characteristic features, contributing
to the correct diagnosis and differentiation between benign and malignant tumors [27, 31, 32]. They also provide information about the location, extent, and spatial relationship of the lesion to adjacent organs, enabling the selection of the appropriate surgical approach. Exclusion of ASMs is important because improper management of these lesions can be dangerous. MRI or spiral CT scans easily identify typical ASMs as unilocular fluid-filled cysts communicating with the thecal sac through sacral bone defects [5, 8, 31, 32]. Retrorectal tumors are traditionally managed by one of four operative approaches: the transsacrococcygeal or posterior, transperineal (interspincteric), transabdominal, and combined abdominosacral approaches [4, 10, 20, 29]. Each has some advantages and disadvantages. A transabdominal procedure allows direct dissection of the mesorectum and minimizes injuries to the ureter and other important pelvic structures. However, a large abdominal incision is required to provide direct vision and enable mobilization in the deep pelvic cavity. Developmental cysts are usually strongly adherent to the coccyx and lower sacrum (Fig. 2) [1, 5, 29, 33], and it can be difficult to sever them at the caudal end through the abdomen. Uhlig et al. [2] reported being unable to achieve complete excision of RTs in four of nine patients via a transabdominal approach. A posterior or transperineal approach is more appropriate for low-lying RTs. The coccyx and, if necessary, part of the lower sacrum can be removed to provide direct access to these ‘‘difficult’’ tumors [29, 32, 34–36]. However, this approach has not been widely accepted because of potential complications such as wound infection, proctocutaneous fistula, and fecal incontinence. In spite of this, a number of authors have advocated this approach for RTs that do not extend above the S2–3 level [4, 6, 31, 32, 35– 37]. Laparoscopic excision of RTs was first described by Sharpe et al. in 1995 [11]. Since then, several authors have reported success with this approach to selected RTs, mostly as case reports [11–25]. In 2013, Duclos et al. [27] and Nedelcu et al. [26] presented short retrospective series of 12 and 9 cases, respectively. We have summarized the results of all published reports, including our series, in Table 2. We excluded the reports of pediatric cases. This summary includes 52 cases, 50 benign and two malignant. In 46 out of 52 cases, the RTs were completely removed via a straight laparoscopic approach. Three open conversions were required because of technical difficulties or intraoperative bleeding [26, 27]. In another three cases, laparoscopic tumor resection was combined with a posterior or perineal approach [19, 22, 24]. Intraoperative complications were reported in only two cases: one rectal injury being successfully suture-repaired without
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Tech Coloproctol Table 2 Published studies reporting the laparoscopic resection of RTs in adults No.
References
Year
1
Sharpe and Van Oppen [11]
1995
No. of cases 1
Tumor diameter (cm)
Pathology
Complication
Approach
5
Dermoid cyst
None
Laparoscopy
2
Melvin [12]
1996
1
3
Schwannoma
NA
Laparoscopy
3
Salameh et al. [13]
2002
1
6
Rectal duplication cyst
None
Laparoscopy
4
K}ohler et al. [14]
2003
1
10
Ganglioneurofibroma
None
Laparoscopy
5
Konstantinidis et al. [15] Lo´pez Cano et al. [16]
2005
2
4, 6
Schwannomas
None
Laparoscopy
2006
1
NA
Tailgut cyst
NA
Laparoscopy
6 7
Chen et al. [17]
2008
1
10
Teratoma
None
Laparoscopy
8
Gunkova et al. [18]
2008
2
10, 5
1 dysontogenetic cyst
None
Laparoscopy
None
Laparoscopy ? perineal
1 epidermoid cysts 9
Palanivelu et al. [19]
2008
1
16
Epidermoid cyst
10
Rao et al. [20]
2010
1
9
Schwannoma
None
Laparoscopy
11
Witherspoon et al. [21]
2010
1
10
Schwannoma
None
Laparoscopy
12
Kye et al. [22]
2011
4
Mean: 5.6 ± 1.1
1 mature teratoma
None
Laparoscopy (N = 3) Laparoscopy ? posterior (N = 1)
1 neurilemmoma 1 fibrous tumor 1 epithelial cyst
13
Lim et al. [23]
2011
1
4
Tailgut cyst
None
14
Marinello et al. [24]
2011
4
11, 10, 7.5, 6.5
2 schwannomas,
Wound infection of sacral incision (N = 1)
1 teratoma
Laparoscopy Laparoscopy (N = 3)
1 solitary fibrous tumor
Residual collection (N = 1)
Laparoscopy ? posterior (N = 1) Laparoscopy
15
Tsutsui et al. [25]
2011
1
6
Teratoma
None
16
Nedelcu [26]
2013
9
Mean: 6.1 ± 2.3
4 schwannomas,
Sciatica (N = 2),
Laparoscopy (N = 8)
2 tailgut cysts,
regional hypoesthesia or paresthesia (N = 2), hematoma (N = 1)
Laparoscopy ? open conversion (N = 1)
1 paraganglioma, 1 meningocele, 1 ganglioneuroma 17
Doclos et al. [27]
2013
12
Mean: 5.6 ± 2.4
10 benign tumors: 2 schwannomas 2 mature teratomas 4 tailgut cysts 1 epidermoid cyst 1 lipoma 2 malignant tumors: 1 ewing sarcoma 1 Colloid sarcoma
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Intraoperative presacral bleeding (N = 1) Intraoperative rectal injury (N = 1) Urinary tract infection (N = 2)
Laparoscopy (N = 10) Laparoscopy ? open conversion (N = 2, including one with ileostomy)
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References
Year
18
Present series
2013
No. of cases 8
Tumor diameter (cm)
Pathology
Complication
Approach
Mean: 8.9 ± 1.7
3 epidermoid cysts
Urinary tract infection (N = 1)
Laparoscopy
2 mature teratomas 2 schwannomas
Constipation (N = 2)
1 leiomyoma NA not available
conversion and one case of intraoperative uncontrollable presacral bleeding necessitating open conversion [27]. Most patients recovered smoothly, minor postoperative complications being observed in 12 cases (23 %) (Table 2). There was no 30-day mortality, and during follow-up ranging from 4 to 79 months, no recurrence occurred [11–27]. Similarly to all previous reports, our study verified the feasibility and safety of a laparoscopic approach to RTs. The advantages of laparoscopy include excellent visibility, deep access, minimal invasiveness, and good cosmetic results; however, a longer operating time may be required. In this study, we did not select patients according to tumor size or location and achieved complete resection (R0) in all cases via a straight laparoscopic approach without open conversion. Our series had a larger mean tumor diameter (8.9 ± 1.7 cm) than the other two short series [26, 27], these differences being statistically significant (p \ 0.05) (Table 2). During our surgical procedures, large cysts filled the operating field, hindering adequate dissection. We therefore partially collapsed cystic lesions by controlled aspiration. In our [6] and other surgeons’ [17, 36] experience with traditional approaches (transsacral or transabdominal), intraoperative rupture of large retrorectal developmental cysts is almost inevitable because of their thin walls, strong adherence, and the narrow surgical space. Abel et al. [36] consider that controlled aspiration to collapse fluid-filled cysts is not contraindicated, even in the presence of infection; the results of our study support this. Including in one case of epidermoid cyst with infection, we found that repeated irrigation of the operating field and effective drainage did not result in either chemical peritonitis or surgical site or wound infection in any of our patients including one case of infected epidermoid cyst. No tumor recurrence occurred in any case after intraoperative rupture of these benign cystic lesions (seven out of eight cases) in this study. According to our previous experience of surgery for 22 benign retrorectal cysts [6], there is no increased risk of tumor recurrence after intraoperative rupture of the lesion. However, because with malignant tumors rupture confers a risk of tumor cell dissemination,
the principles of en bloc resection should always be followed for malignant RTs. With regard to location, the average distance from the inferior end of the tumor to the anal verge was 4.1 cm (range 2–7 cm) in this series. The inferior parts of two epidermoid cysts (cases 3 and 7) were buried beneath the pelvic floor muscles. Preoperatively, we anticipated difficulty in dealing with them simply by laparoscopy and planned a combined posterior approach. However, after reaching the pelvic floor muscles during surgery, continuous dissecting along the plane between the cystic capsule and the adherent levator ani muscles achieved entry to the ischiorectal fossa, enabling the removal of the entire lesion from tits last attachment to the levator ani muscles, as shown in Fig. 1d. R0 resection was achieved in both cases with operating times of 180 and 115 min and with minimal blood loss. The above studies show that a laparoscopic procedure is a valid alternative to traditional surgical approaches to RTs. Nedelcu et al. [26] recommended a laparoscopic approach for potentially benign RTs located above S3 with diameters \ 6 cm and not involving adjacent organs and bones. Nevertheless, in this study, we showed that benign cystic lesions larger than 6 cm and lower than S3 can also be safely removed via a straight laparoscopic approach. Previous studies have shown that achieving R0 resection is much more difficult in malignant than in benign RTs, whatever surgical approach is selected [31, 38, 39]. As a result, local recurrence occurs more frequently after resection of malignant than of benign tumors [1, 38]. So far, no report has described the laparoscopic approach for malignant RTs. Duclos et al. [27] reported the only two malignant RTs that have been subjected to laparoscopic procedures to date, both having been misdiagnosed as benign lesions preoperatively. One, a retrorectal Ewing sarcoma, required an open conversion because of technical difficulties. The other, a retrorectal colloid sarcoma, was incompletely resected and re-operated on with an open technique after neoadjuvant chemoradiation. Because in cases of malignant disease a more aggressive procedure is required in order to achieve radical resection with tumor-
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free margins without entering the lesion, the likelihood of malignancy must be assessed preoperatively. A combined approach is often mandatory; a multidisciplinary team including general surgeons, orthopedists, neurosurgeons, and plastic surgeons likely increases the chance of achieving radical resection [1, 5, 40]. As for ASMs, a rare congenital malformation, most authors recommend a posterior approach, which allows identification and ligation of the communicating stalk with a low risk of sepsis and neural damage [1, 5, 41, 42]. We therefore do not suggest the straight laparoscopic approach for this condition. However, on the basis of other authors’ experiences with a combined traditional approach (e.g., combined transabdominal/transsacral) [1, 5], we presume that a laparoscopic approach could be combined with a posterior approach, if necessary, to facilitate the amputation of the sac and removal of the combined presacral lesions (since, as described by Jao et al. [1], ASMs can occur in combination with other presacral tumors such as developmental cysts or lipoma). Further studies are needed to confirm this speculation. We acknowledge that the current series is small, retrospective, and from a single unit. Our results may not reflect outcomes that would be achieved using the same technique in other centers. Future studies should include more cases to avoid bias. A prospective cohort study comparing laparoscopic and traditional approaches would produce more valid results.
Conclusions Our results support the safety and feasibility of a laparoscopic approach to benign RTs with the benefits of minimally invasive surgery. In addition, large benign RTs, including those extending below the pelvic floor muscles, can be entirely removed via a straight laparoscopic approach with the deep access and excellent visualization provided by this technique, and by collapsing large cystic lesions by controlled aspiration. We believe that a laparoscopic approach may become an ideal alternative to traditional approaches for treating RTs. Meanwhile, it can be combined with a posterior approach if necessary, for example, to remove extensive benign lesions involving the sacrum. Acknowledgments The authors express their gratitude to Professor Bo Shen, who was extremely helpful and offered invaluable assistance and guidance. Conflict of interest Drs. Jiao-lin Zhou, Bin Wu, Yi Xiao, Guo-le Lin, and Hui-zhong Qiu have undergone training programs for laparoscopic surgery in the training centers of Ethicon and Covidien, and
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these companies having paid for this training support. Drs. Wen-ze Wang and Guan-nan Zhang have no conflict of interests to disclose.
References 1. Jao SW, Beart RW, Spencer RJ, Reiman HM, Ilstrup DM (1985) Retrorectal tumors. Dis Colon Rectum 28:644–652 2. Uhlig BE, Johnson RL (1975) Presacral tumors and cysts in adults. Dis Colon Rectum 18:581–589 3. McCune WS (1964) Management of sacrococcygeal tumors. Ann Surg 159:911–918 4. Glasgow SC, Birnbaum EH, Lowney JK et al (2005) Retrorectal tumors: a diagnostic and therapeutic challenge. Dis Colon Rectum 48:1581–1587 5. Hobson KG, Ghaemmaghami V, Roe JP, Goodnight JE, Khatri VP (2005) Tumors of the retrorectal space. Dis Colon Rectum 48:1964–1974 6. Zhou JL, Qiu HZ (2010) Management of presacral developmental cysts: experience of 22 cases. Zhonghua Wai Ke Za Zhi 48:284–287 7. Sobrado CW, Mester M, Simonsen OS, Justo CR, deAbreu JN, Habr-Gama A (1996) Retrorectal tumors complicating pregnancy. Report of two cases. Dis Colon Rectum 39:1176–1179 8. Dahan H, Arrive L, Wendum D, Docou le Pointe H, Djouhri H, Tubiana JM (2001) Retrorectal developmental cysts in adults: clinical and radiologic-histopathologic review, differential diagnosis, and treatment. Radiographics 21:575–584 9. Kalady MF, Ludwig KA (2004) Presacral developmental cysts. Semin Colon Rectal Surg 15:12–18 10. Nair RR, Shoukrey MN, Whitlow B (2007) Presacral dermoid: now you see it now you don’t. Gynecol Surg 4:229–231 11. Sharpe LA, Van Oppen DJA (1995) Laparoscopic removal of a benign pelvic retroperitoneal dermoid cyst. J Am Assoc Gynecol Laparosc 2:223–226 12. Melvin WS (1996) Laparoscopic resection of a pelvic schwannoma. Surg Laparosc Endosc 6:489–491 13. Salameh JR, Votanopoulos KI, Hilal RE et al (2002) Rectal duplication cyst in an adult: the laparoscopic approach. J Laparoendosc Adv Surg Tech A 12:453–456 14. Kohler C, Kuhne-Heid R, Klemm P, Tozzi R, Schneider A (2003) Resection of presacral ganglioneurofibroma by laparoscopy. Surg Endosc 17:1499 15. Konstantinidis K, Theodoropoulos GE, Sambalis G et al (2005) Laparoscopic resection of presacral schwannomas. JSLS 15:302–304 16. Lopez Cano M, Vilallonga R, Espin Basany E, Sanchez Garcia JL, Lozoya Trujillo R, Armengol Carrasco M (2006) Retrorectal tumors in adults. Experience in five cases. Cir Esp 80:334–336 17. Chen Y, Xu H, Li Y et al (2008) Laparoscopic resection of presacral teratomas. J Minim Invasive Gynecol 15:649–651 18. Gunkova P, Martinek L, Dostalik J, Gunka I, Vavra P, Mazur M (2008) Laparoscopic approach to retrorectal cyst. World J Gastroenterol WJG 14:6581–6583 19. Palanivelu C, Rangarajan M, Senthilkumar R, Madankumar MV, Annapoorni S (2008) Laparoscopic and perineal excision of an infected ‘‘dumb-bell’’ shaped retrorectal epidermoid cyst. J Laparoendoscopic Adv Surg Tech 8:88–92 20. Rao M, Sagar P, Duff S, Hulme-Moir M, Brayshaw I (2010) Laparoscopic excision of a retrorectal schwannoma. Tech Coloproctol 14:353–355 21. Witherspoon P, Armitage J, Gatt M, Sagar PM (2010) Laparoscopic excision of retrorectal schwannoma. Dis Colon Rectum 53:101–103
Tech Coloproctol 22. Kye BH, Kim HJ, Cho HM, Chin HM, Kim JG (2011) Clinicopathological features of retrorectal tumors in adults: 9 years of experience in a single institution. J Korean Surg Soc 81:122–127 23. Lim SW, Huh JW, Kim YJ, Kim HR (2011) Laparoscopyassisted resection of tailgut cysts: report of a case. Case Rep Gastroenterol 5:22–27 24. Marinello FG, Targarona EM, Luppi CR, Boguna I, Molet J, Trias M (2011) Laparoscopic approach to retrorectal tumors: review of the literature and report of 4 cases. Surg Laparosc Endosc Percutan Tech 21:10–13 25. Tsutsui A, Nakamura T, Mitomi H et al (2011) Successful laparoscopic resection of a sacrococcygeal teratoma in an adult: report of a case. Surg Today 41:572–575 26. Nedelcu M, Andreica A, Skalli M et al (2013) Laparoscopic approach for retrorectal tumors. Surg Endosc 27:4177–4183 27. Duclos J, Maggiori L, Zappa M, Ferron M, Panis Y (2013) Laparoscopic resection of retrorectal tumors: a feasibility study in 12 consecutive patients. Surg Endosc PMID: 24263459 28. Coco C, Manno A, Mattana C et al (2008) Congenital tumors of the retrorectal space in the adult: report of two cases and review of the literature. Tumori 94:602–607 29. Bohm B, Milsom JW, Fazio VW, Lavery IC, Church JM, Oakley JR (1993) Our approach to the management of congenital presacral tumors in adults. Int J Colorectal Dis 8:134–138 30. Lu NH, Tseng MJ (2010) Laparoscopic management of tailgut cyst: case report and review of literature. J Minim Invasive Gynecol 17:802–804 31. Macafee DA, Sagar PM, El-Khoury T, Hyland R (2012) Retrorectal tumours: optimization of surgical approach and outcome. Colorectal Dis 14:1411–1417
32. Chereau N, Lefevre JH, Meurette G et al (2013) Surgical resection of retrorectal tumours in adults: long-term results in 47 patients. Colorectal Dis 15:e476–e482 33. Miles RM, Stewart GS Jr (1974) Sacrococcygeal teratomas in adult. Ann Surg 179:676–683 34. Geelhoed GW, Kotz HL (1983) A posterior approach to the presacral mass. World J Surg 7:620–628 35. Pena A, Hong A (2004) The posterior sagittal trans-sphincteric approach. Acta Chir Iugosl 51:11–21 36. Abel ME, Nelson R, Prasad ML, Pearl RK, Orsay CP, Abcarian H (1985) Parasacrococcygeal approach for the resection of retrorectal developmental cysts. Dis Colon Rectum 28:855–858 37. Buchs N, Taylor S, Roche B (2007) The posterior approach for low retrorectal tumors in adults. Int J Colorectal Dis 22:381–385 38. Lev-Chelouche D, Gutman M, Goldman G et al (2003) Presacral tumors: a practical classification and treatment of a unique and heterogeneous group of diseases. Surgery 133:473–478 39. Woodfield JC, Chalmers AG, Phillips N, Sagar PM (2008) Algorithms for the surgical management of retrorectal tumours. Br J Surg 95:214–221 40. Dozois EJ, Jacofsky DJ, Billings BJ et al (2011) Surgical approach and oncologic outcomes following multidisciplinary management of retrorectal sarcomas. Ann Surg Oncol 18:983–988 41. Massimi L, Calisti A, Koutzoglou M, Di Rocco C (2003) Giant anterior sacral meningocele and posterior sagittal approach. Childs Nerv Syst 19:722–728 42. Villarejo F, Scavone C, Blazquez MG et al (1983) Anterior sacral meningocele: review of the literature. Surg Neurol 19:57–71
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ORIGINAL ARTICLE
A laparoscopic approach to benign retrorectal tumors J. L. Zhou • B. Wu • Y. Xiao • G. L. Lin W. Z. Wang • G. N. Zhang • H. Z. Qiu
•
Received: 5 January 2014 / Accepted: 25 March 2014 Ó Springer-Verlag Italia 2014
Abstract Background Retrorectal tumors (RTs) are rare in adults. Their surgical excision is often difficult because of their anatomic location. The aim of this study was to evaluate the results of straight laparoscopic resection of RTs in our institution. Methods Eight patients (six women and two men) with benign RTs were treated by laparoscopic resection in our tertiary care center between September 2012 and June 2013. Exclusion criteria included malignant tumors, lesions with fistula formation, and anterior sacral meningoceles. Clinical data, imaging features, operative details, pathological results, and treatment outcomes were reviewed and analyzed. Results Eight cases of benign RT with an average diameter of 8.9 ± 1.7 cm were treated by a straight laparoscopic procedure. The mean operative time was 122 ± 36 min, and the average intraoperative blood loss was 46 ± 33 ml. The median postoperative stay was 5 days (range 3–8 days), and all patients were discharged without serious complications. During a median follow-up of 11 months, no tumor recurrence was observed.
J. L. Zhou and B. Wu have contributed equally to this study. J. L. Zhou B. Wu Y. Xiao G. L. Lin G. N. Zhang H. Z. Qiu (&) Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Dong Dan, Dong Cheng District, Beijing 100730, China e-mail: [email protected] W. Z. Wang Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Beijing, China
Conclusions In our experience, a laparoscopic approach is safe for removing benign tumors in the retrorectal space. This approach may provide access to this difficult-to-reach space and has the advantages of allowing excellent visualization, meticulous dissection, less morbidities, and fast recuperation. Keywords Benign
Laparoscopy Adults Retrorectal tumor
Introduction Retrorectal tumors (RTs), a group of heterogeneous and uncommon lesions, develop in the retrorectal space. This potential space, which lies anterior to the sacrum and coccyx and posterior to the rectum, is also called the presacral space [1, 2]. RTs are rare in adults, with an estimated incidence of one in 40,000–63,000 admissions to large referral centers [1, 3, 4]. Benign RTs may have potential for malignant degeneration [5, 6]. After enlarging, they may interfere with the pelvic outlet, causing difficult or incomplete evacuation of stools, and can even lead to obstructive dystocia in women of childbearing age [2, 6, 7]. Developmental cysts are associated with a risk of secondary infection and may form fistulas to the sacrococcygeal skin or into the rectum [6, 8]. It is commonly believed that RTs should be surgically removed once diagnosed [1, 5, 9, 10]. Their anatomic location makes RTs hard to reach and so they have traditionally been accessed through transsacral, transabdominal, or a combined abdominosacral route. Each approach has advantages and disadvantages. A laparoscopic approach to RTs was first reported in the 1990s. Recently, successful laparoscopic resections of
123
123
No 3 None 20 F 8
BMI body mass index, DRE digital rectal examination, M male, F female, CT computed tomography, MRI magnetic resonance imaging, US ultrasound
80 US ? CT 9.4 9 4.1 9 3.1 ? None Laparoscopic oophorocystectomy None Leiomyoma 20
F 7
43
No 5 None 40 115 US ? MRI 9.3 9 9.0 9 7.2 ? None None None Epidermoid cyst 19.2
F 6
27
No 5 None 50 150 US ? CT 9.0 9 9.0 9 8.0 ? None None None Mature teratoma 22.5
M 5
15
No 7 None 30 150 CT 10.0 9 5.4 9 4.5 ? Hypertension, hyperlipoidemia, and Cushing’s syndrome Laparotomy for mature teratoma of bladder None Mature teratoma 20.8
F 4
23
No 5 None 30 85 US ? CT 7.0 9 6.6 9 4.6 ? None Cesarean section None Epidermoid cyst 21.2
F 3
29
No 3 None 20 180 MRI 11.8 9 8.8 9 8.0 ? None Open hysteromyomectomy None Epidermoid cyst 21.8
F 2
47
No 4 None 120 95 US ? CT 6.3 9 6.2 9 5.8 Hypertension
M
63
26
Schwannoma
Lower abdominal pain
Laparoscopic exploration
–
No 5 None 60 120 CT 8.0 9 7.5 9 7.1 ? Hypertension and coronary heart disease None None Schwannoma 23.9
Operating time (min) Diagnosis Size (cm) DRE Comorbidities Previous abdominal surgery Symptom Pathologic diagnosis
1
All operations were performed by two colorectal surgeons with skill in laparoscopic colorectal surgeries. The patient was placed in a steep Trendelenburg position. Five ports (two 10-mm and three 5-mm ports) placed in a similar pattern as that for laparoscopic rectal cancer resection were used. In most cases, dissection began from the right side of
BMI
Surgical technique
Age (years)
Results are presented as median values or mean ± standard deviation (SD) for continuous variables. The independentsamples t test was used to assess tumor size differences between this and two other series. All tests for statistical significance were two-tailed, the level of significance being set at p \ 0.05. All data were analyzed using SPSS software (version 19.0 for Windows, SPSS, Chicago, IL, USA).
Sex
Statistical analysis
No.
In the period from September 2012 to June 2013, eight patients with benign RTs underwent straight laparoscopic resection performed by two experienced colorectal surgeons at our tertiary care center. The preoperative diagnoses were made on the basis of clinical features and imaging including ultrasound (US), computerized tomography (CT) scan, or magnetic resonance imaging (MRI). During the study period, laparoscopic approaches were used for retrorectal lesions identified as benign regardless of tumor size or extent of the lesion. Lesions identified as malignant tumors, benign lesions with severe infection or fistula formation, and anterior sacral meningoceles (ASM) were excluded. Data including the patients’ sex, age, clinical presentation, physical examination, imaging characteristics, final pathological diagnosis, operative details, intra- and postoperative complications, postoperative length of stay, mortality, and recurrence rates were recorded and summarized (Table 1).
Table 1 Description of retrorectal tumor cases treated by the laparoscopic excision including patient demographics and clinical variables
Materials and methods
Intraoperative blood loss (ml)
Complications
Postoperative length of stay (days)
benign RTs have been reported, mostly as initial attempts [11–27]. In this study, we reviewed eight cases of benign RT treated with laparoscopic surgery. We examined patient characteristics, operative details, perioperative morbidities, and outcomes to assess the feasibility, safety, and oncological outcome (in terms of incomplete excision or tumor recurrence) of the laparoscopic approach. Selected aspects of the diagnosis of RTs and surgical procedures are also discussed.
75
Recurrence
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the upper mesorectum above the sacral promontory. After the right retroperitoneum had been opened with a harmonic scalpel, the flimsy areolar tissue in the plane between the visceral mesorectal fascia and the parietal pelvic fascia was identified. Dissection proceeded medially and downward along this plane to mobilize the rectum from the retrorectal space. Care was taken to identify and safeguard the ureters and pelvic autonomic nerves. Left-sided tumors were accessed from the left. For large lesions, opening the retroperitoneum on both sides provided better exposure. With the rectum retracted upward and anteriorly, dissection continued downward until the lesion was revealed (Fig. 1a). The tumor was carefully divided from the pelvic side wall and adjacent structures along its capsule (Fig. 1b). It was sometimes difficult to distinguish the borders of secondarily infected retrorectal cysts. The correct plane was carefully identified to prevent cutting into the mesorectum and causing bleeding or injuring the rectal wall. When a cystic lesion occupied too much of the working space, it was partially collapsed by aspiration to allow dissection to continue. The collapsed cyst was moved aside to reveal its remaining attachments, greatly
facilitating further dissection (Fig. 1c). Retrorectal developmental cysts usually have a dense attachment to the coccyx and anterior surface of the lower sacrum (Fig. 2) and may extend into or below the pelvic floor muscles. In such cases, dissection was continued downward close to the tumor capsule. Attachment to the levator ani muscles was severed, and the inferior end of the lesion dissected off until the entire lesion could be removed (Fig. 1c, d), taking care to distinguish the muscle fibers of the levator ani from those of the rectal wall so as not to injure the latter. The specimen was extracted using a laparoscopic retrieval bag (Endocatch Gold 10 mm; Surgical Corporation, Norwalk, CT, USA) via a prolonged trocar incision and the retrorectal space adequately irrigated. A drain was placed in the surgical anatomic space and brought out through a trocar incision. Before surgery, all patients were fully informed about the procedure and potential alternatives, risks, and complications, and provided written informed consent. Bowel preparation with polyethylene glycol electrolytes powder was performed the afternoon before the operation and a single dose of prophylactic antibiotic given just prior
Fig. 1 Details of laparoscopic resection of a retrorectal epidermoid cyst (case 7). a The top of the retrorectal cyst (arrow 1) was revealed from behind the mesorectum (arrow 2) by harmonic dissection between the posterior mesorectal fascia and the presacral fascia. b Care was given to dissect the cyst (arrow 1) along its capsule to prevent injury to the rectal wall (arrow 2). c Dissection continued
downward between the collapsed cyst (encircled by the curved line) and the musculature of the pelvic floor (white arrows) to enter the ischiorectal space (black arrow), to reach the lower end of the cyst. d Finally, the collapsed cyst (black arrow) was cut from its attachment to the levator ani muscle (white arrow)
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Fig. 2 Sagittal T2-weighted MR image showing the retrorectal epidermoid cyst in case 7, which is adherent to the coccyx and lower sacrum and extends below the levator ani muscle
to induction of anesthesia. Postoperative interventions included intravenous fluid and on-demand analgesia. Early mobilization was encouraged. Oral water intake started the day after surgery, and a liquid diet was initiated once the patient passed flatus or had a bowel movement. Patients were discharged according to the hospital’s discharge criteria. Follow-up Follow-up was conducted by interview in the outpatient clinic and by telephone. All patients were required to return to the outpatient clinic 1 and 6 months postoperatively, to attend for long-term follow-up thereafter, and to return to our hospital whenever there was any suspicion of disease recurrence. Digital rectal examination (DRE) and pelvic US were performed routinely to detect recurrence. The long-term complications and rate of recurrence were recorded at follow-up.
Results The series of eight patients consisted of six women and two men with a median age of 36 years (range 15–75 years). The diagnoses on final pathological examinations were three epidermoid cysts (including one infected cyst), two mature teratomas, two schwannomas, and one leiomyoma; all were benign (Table 1).
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Seven patients were asymptomatic, their lesions having been detected incidentally during imaging studies performed for other purposes. One 63-year-old woman with a schwannoma (patient 2) had noticed lower abdominal pain and fullness 1 month before discovery of the tumor. On physical examination, a retrorectal mass (behind or beside the rectum) was detected by DRE in seven patients. The mean distance, measured by DRE, from the lower end of the mass to the anal verge was 4.1 ± 1.7 cm. In six patients, the upper end of the lesion could not be reached on DRE. In one patient with schwannoma, the tumor could not be reached on DRE but it was detectable as an immobile round hard pelvic mass by abdominal palpation. Pelvic US was performed in five patients. Six patients underwent spiral CT scan and another two underwent MRI, which clearly defined the location of the tumors and their relationships with adjacent structures. All tumors were shown to be well-defined retrorectal soft-tissue masses that did not invade of adjacent organs. According to the imaging studies, the largest tumors ranged from 6.3 to 11.8 cm in diameter (mean 8.9 ± 1.7 cm). Straight laparoscopy was completed in all patients, neither conversion to open surgery nor combination with other approaches being required. All five developmental cysts (three epidermoid cysts and two mature teratomas) were decompressed during surgery to improve exposure and facilitate dissection, either by intentionally puncturing them followed by aspiration with a suction device or after accidental rupture during dissection. Any semisolid material spilled from the cysts was removed by suction and repeated lavage to prevent postoperative complications such as peritonitis, adhesion formation, and wound complications. Two of the epidermoid cysts (patients 3 and 7) extended beneath the pelvic floor muscles, necessitating the continuation of dissection downward between the cystic capsule and levator ani muscles to enter the ischiorectal space. Macroscopically complete tumor removal was achieved in all eight cases. No injury to the rectal wall or other pelvic viscera occurred. The mean operating time was 122 ± 36 min, and intraoperative blood loss was insignificant (mean 46 ± 33 ml). Median postoperative length of stay was 5 days (range 3–8 days). Postoperatively, urinary tract infection occurred on day 5 in one patient (patient 5) with the comorbidity of ectopic adrenocorticotropic hormone syndrome. Antibiotics were administered with relief of symptoms within 2 days. In the other seven patients, postoperative fever of \38.5 °C was observed only within 48 h of surgery. No bowel damage, postoperative bowel obstruction, surgical site infection, or wound complication occurred; there was no perioperative mortality. During a median follow-up of 11 months (range 4–14 months), no residual lesion or tumor recurrence was
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observed. Two patients complained that they felt no urge to defecate and had constipation; these symptoms improved with laxatives and increased dietary fiber. No other longterm complication was observed.
Discussion The retrorectal space is an area of development of multiple embryologic structures; a heterogeneous group of benign and malignant tumors can arise from their remnants [5, 28]. Most RTs are benign, and these benign tumors are most often developmental cysts (epidermoid, dermoid and tailgut cysts, and benign teratomas) and schwannomas. According to the previous reports, RTs are rare [1, 3]. However, because many remain clinically silent, the true prevalence of these lesions is unknown [4]. This study included eight patients with benign RTs treated at our institution within a period of 10 months. Seven were asymptomatic, their lesions being detected accidentally during imaging for other reasons. With the increasing performance of imaging studies during health checks, more asymptomatic cases will likely be uncovered. Symptoms of benign RTs are usually nonspecific or related to local mass effect [8]. Patients may complain of pain, a sensation of downward pressure in the pelvis or rectum, or a change in bowel habits [29]. Patients with large lesions may present with difficult or incomplete defecation or even dysuria. Among women of childbearing age, such masses occasionally obstruct labor, necessitating Cesarean section [2, 5]. It is generally considered that RTs should be surgically removed once diagnosed, even when asymptomatic and benign [1, 5, 9, 10]. Almost all RTs can be palpated on DRE [1], which can determine their size, location, consistency, and relationship with the rectal wall and surrounding structures. DRE is therefore the simplest, most efficient method for identifying these lesions. However, because of their soft texture and the smoothness of the overlying rectal mucosa, they can escape detection on DRE. Thus, careful palpation by sweeping the examining finger across the mucosal surface is necessary. Retrorectal tumors are readily revealed by transabdominal US. However, inexperienced sonographers can misdiagnose retrorectal cysts as adnexal lesions, especially when the ovaries are not visualized precisely. In this series, case 2 was initially misdiagnosed as having a left ovarian tumor and underwent an unfruitful laparoscopic exploration in the gynecology department. Similar cases have been reported by other authors [10, 30]. Spiral CT scan and MRI, especially sagittal plane images (Fig. 2), are very helpful in defining RTs. They demonstrate these tumors’ characteristic features, contributing
to the correct diagnosis and differentiation between benign and malignant tumors [27, 31, 32]. They also provide information about the location, extent, and spatial relationship of the lesion to adjacent organs, enabling the selection of the appropriate surgical approach. Exclusion of ASMs is important because improper management of these lesions can be dangerous. MRI or spiral CT scans easily identify typical ASMs as unilocular fluid-filled cysts communicating with the thecal sac through sacral bone defects [5, 8, 31, 32]. Retrorectal tumors are traditionally managed by one of four operative approaches: the transsacrococcygeal or posterior, transperineal (interspincteric), transabdominal, and combined abdominosacral approaches [4, 10, 20, 29]. Each has some advantages and disadvantages. A transabdominal procedure allows direct dissection of the mesorectum and minimizes injuries to the ureter and other important pelvic structures. However, a large abdominal incision is required to provide direct vision and enable mobilization in the deep pelvic cavity. Developmental cysts are usually strongly adherent to the coccyx and lower sacrum (Fig. 2) [1, 5, 29, 33], and it can be difficult to sever them at the caudal end through the abdomen. Uhlig et al. [2] reported being unable to achieve complete excision of RTs in four of nine patients via a transabdominal approach. A posterior or transperineal approach is more appropriate for low-lying RTs. The coccyx and, if necessary, part of the lower sacrum can be removed to provide direct access to these ‘‘difficult’’ tumors [29, 32, 34–36]. However, this approach has not been widely accepted because of potential complications such as wound infection, proctocutaneous fistula, and fecal incontinence. In spite of this, a number of authors have advocated this approach for RTs that do not extend above the S2–3 level [4, 6, 31, 32, 35– 37]. Laparoscopic excision of RTs was first described by Sharpe et al. in 1995 [11]. Since then, several authors have reported success with this approach to selected RTs, mostly as case reports [11–25]. In 2013, Duclos et al. [27] and Nedelcu et al. [26] presented short retrospective series of 12 and 9 cases, respectively. We have summarized the results of all published reports, including our series, in Table 2. We excluded the reports of pediatric cases. This summary includes 52 cases, 50 benign and two malignant. In 46 out of 52 cases, the RTs were completely removed via a straight laparoscopic approach. Three open conversions were required because of technical difficulties or intraoperative bleeding [26, 27]. In another three cases, laparoscopic tumor resection was combined with a posterior or perineal approach [19, 22, 24]. Intraoperative complications were reported in only two cases: one rectal injury being successfully suture-repaired without
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Tech Coloproctol Table 2 Published studies reporting the laparoscopic resection of RTs in adults No.
References
Year
1
Sharpe and Van Oppen [11]
1995
No. of cases 1
Tumor diameter (cm)
Pathology
Complication
Approach
5
Dermoid cyst
None
Laparoscopy
2
Melvin [12]
1996
1
3
Schwannoma
NA
Laparoscopy
3
Salameh et al. [13]
2002
1
6
Rectal duplication cyst
None
Laparoscopy
4
K}ohler et al. [14]
2003
1
10
Ganglioneurofibroma
None
Laparoscopy
5
Konstantinidis et al. [15] Lo´pez Cano et al. [16]
2005
2
4, 6
Schwannomas
None
Laparoscopy
2006
1
NA
Tailgut cyst
NA
Laparoscopy
6 7
Chen et al. [17]
2008
1
10
Teratoma
None
Laparoscopy
8
Gunkova et al. [18]
2008
2
10, 5
1 dysontogenetic cyst
None
Laparoscopy
None
Laparoscopy ? perineal
1 epidermoid cysts 9
Palanivelu et al. [19]
2008
1
16
Epidermoid cyst
10
Rao et al. [20]
2010
1
9
Schwannoma
None
Laparoscopy
11
Witherspoon et al. [21]
2010
1
10
Schwannoma
None
Laparoscopy
12
Kye et al. [22]
2011
4
Mean: 5.6 ± 1.1
1 mature teratoma
None
Laparoscopy (N = 3) Laparoscopy ? posterior (N = 1)
1 neurilemmoma 1 fibrous tumor 1 epithelial cyst
13
Lim et al. [23]
2011
1
4
Tailgut cyst
None
14
Marinello et al. [24]
2011
4
11, 10, 7.5, 6.5
2 schwannomas,
Wound infection of sacral incision (N = 1)
1 teratoma
Laparoscopy Laparoscopy (N = 3)
1 solitary fibrous tumor
Residual collection (N = 1)
Laparoscopy ? posterior (N = 1) Laparoscopy
15
Tsutsui et al. [25]
2011
1
6
Teratoma
None
16
Nedelcu [26]
2013
9
Mean: 6.1 ± 2.3
4 schwannomas,
Sciatica (N = 2),
Laparoscopy (N = 8)
2 tailgut cysts,
regional hypoesthesia or paresthesia (N = 2), hematoma (N = 1)
Laparoscopy ? open conversion (N = 1)
1 paraganglioma, 1 meningocele, 1 ganglioneuroma 17
Doclos et al. [27]
2013
12
Mean: 5.6 ± 2.4
10 benign tumors: 2 schwannomas 2 mature teratomas 4 tailgut cysts 1 epidermoid cyst 1 lipoma 2 malignant tumors: 1 ewing sarcoma 1 Colloid sarcoma
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Intraoperative presacral bleeding (N = 1) Intraoperative rectal injury (N = 1) Urinary tract infection (N = 2)
Laparoscopy (N = 10) Laparoscopy ? open conversion (N = 2, including one with ileostomy)
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References
Year
18
Present series
2013
No. of cases 8
Tumor diameter (cm)
Pathology
Complication
Approach
Mean: 8.9 ± 1.7
3 epidermoid cysts
Urinary tract infection (N = 1)
Laparoscopy
2 mature teratomas 2 schwannomas
Constipation (N = 2)
1 leiomyoma NA not available
conversion and one case of intraoperative uncontrollable presacral bleeding necessitating open conversion [27]. Most patients recovered smoothly, minor postoperative complications being observed in 12 cases (23 %) (Table 2). There was no 30-day mortality, and during follow-up ranging from 4 to 79 months, no recurrence occurred [11–27]. Similarly to all previous reports, our study verified the feasibility and safety of a laparoscopic approach to RTs. The advantages of laparoscopy include excellent visibility, deep access, minimal invasiveness, and good cosmetic results; however, a longer operating time may be required. In this study, we did not select patients according to tumor size or location and achieved complete resection (R0) in all cases via a straight laparoscopic approach without open conversion. Our series had a larger mean tumor diameter (8.9 ± 1.7 cm) than the other two short series [26, 27], these differences being statistically significant (p \ 0.05) (Table 2). During our surgical procedures, large cysts filled the operating field, hindering adequate dissection. We therefore partially collapsed cystic lesions by controlled aspiration. In our [6] and other surgeons’ [17, 36] experience with traditional approaches (transsacral or transabdominal), intraoperative rupture of large retrorectal developmental cysts is almost inevitable because of their thin walls, strong adherence, and the narrow surgical space. Abel et al. [36] consider that controlled aspiration to collapse fluid-filled cysts is not contraindicated, even in the presence of infection; the results of our study support this. Including in one case of epidermoid cyst with infection, we found that repeated irrigation of the operating field and effective drainage did not result in either chemical peritonitis or surgical site or wound infection in any of our patients including one case of infected epidermoid cyst. No tumor recurrence occurred in any case after intraoperative rupture of these benign cystic lesions (seven out of eight cases) in this study. According to our previous experience of surgery for 22 benign retrorectal cysts [6], there is no increased risk of tumor recurrence after intraoperative rupture of the lesion. However, because with malignant tumors rupture confers a risk of tumor cell dissemination,
the principles of en bloc resection should always be followed for malignant RTs. With regard to location, the average distance from the inferior end of the tumor to the anal verge was 4.1 cm (range 2–7 cm) in this series. The inferior parts of two epidermoid cysts (cases 3 and 7) were buried beneath the pelvic floor muscles. Preoperatively, we anticipated difficulty in dealing with them simply by laparoscopy and planned a combined posterior approach. However, after reaching the pelvic floor muscles during surgery, continuous dissecting along the plane between the cystic capsule and the adherent levator ani muscles achieved entry to the ischiorectal fossa, enabling the removal of the entire lesion from tits last attachment to the levator ani muscles, as shown in Fig. 1d. R0 resection was achieved in both cases with operating times of 180 and 115 min and with minimal blood loss. The above studies show that a laparoscopic procedure is a valid alternative to traditional surgical approaches to RTs. Nedelcu et al. [26] recommended a laparoscopic approach for potentially benign RTs located above S3 with diameters \ 6 cm and not involving adjacent organs and bones. Nevertheless, in this study, we showed that benign cystic lesions larger than 6 cm and lower than S3 can also be safely removed via a straight laparoscopic approach. Previous studies have shown that achieving R0 resection is much more difficult in malignant than in benign RTs, whatever surgical approach is selected [31, 38, 39]. As a result, local recurrence occurs more frequently after resection of malignant than of benign tumors [1, 38]. So far, no report has described the laparoscopic approach for malignant RTs. Duclos et al. [27] reported the only two malignant RTs that have been subjected to laparoscopic procedures to date, both having been misdiagnosed as benign lesions preoperatively. One, a retrorectal Ewing sarcoma, required an open conversion because of technical difficulties. The other, a retrorectal colloid sarcoma, was incompletely resected and re-operated on with an open technique after neoadjuvant chemoradiation. Because in cases of malignant disease a more aggressive procedure is required in order to achieve radical resection with tumor-
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free margins without entering the lesion, the likelihood of malignancy must be assessed preoperatively. A combined approach is often mandatory; a multidisciplinary team including general surgeons, orthopedists, neurosurgeons, and plastic surgeons likely increases the chance of achieving radical resection [1, 5, 40]. As for ASMs, a rare congenital malformation, most authors recommend a posterior approach, which allows identification and ligation of the communicating stalk with a low risk of sepsis and neural damage [1, 5, 41, 42]. We therefore do not suggest the straight laparoscopic approach for this condition. However, on the basis of other authors’ experiences with a combined traditional approach (e.g., combined transabdominal/transsacral) [1, 5], we presume that a laparoscopic approach could be combined with a posterior approach, if necessary, to facilitate the amputation of the sac and removal of the combined presacral lesions (since, as described by Jao et al. [1], ASMs can occur in combination with other presacral tumors such as developmental cysts or lipoma). Further studies are needed to confirm this speculation. We acknowledge that the current series is small, retrospective, and from a single unit. Our results may not reflect outcomes that would be achieved using the same technique in other centers. Future studies should include more cases to avoid bias. A prospective cohort study comparing laparoscopic and traditional approaches would produce more valid results.
Conclusions Our results support the safety and feasibility of a laparoscopic approach to benign RTs with the benefits of minimally invasive surgery. In addition, large benign RTs, including those extending below the pelvic floor muscles, can be entirely removed via a straight laparoscopic approach with the deep access and excellent visualization provided by this technique, and by collapsing large cystic lesions by controlled aspiration. We believe that a laparoscopic approach may become an ideal alternative to traditional approaches for treating RTs. Meanwhile, it can be combined with a posterior approach if necessary, for example, to remove extensive benign lesions involving the sacrum. Acknowledgments The authors express their gratitude to Professor Bo Shen, who was extremely helpful and offered invaluable assistance and guidance. Conflict of interest Drs. Jiao-lin Zhou, Bin Wu, Yi Xiao, Guo-le Lin, and Hui-zhong Qiu have undergone training programs for laparoscopic surgery in the training centers of Ethicon and Covidien, and
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these companies having paid for this training support. Drs. Wen-ze Wang and Guan-nan Zhang have no conflict of interests to disclose.
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