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ORIGINAL ARTICLE
Year : 2019  |  Volume : 15  |  Issue : 1  |  Page : 132-137

A clinical and surgical challenge: Retrorectal tumors


1 Department of General Surgery, Health Science University, Umraniye Education and Research Hospital, Umraniye, Istanbul, Turkey
2 Department of Radiology, Health Science University, Umraniye Education and Research Hospital, Umraniye, Istanbul, Turkey

Date of Web Publication13-Mar-2019

Correspondence Address:
Dr. Fatih Basak
Department of General Surgery, Umraniye Education and Research Hospital, Istanbul
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.183192

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 > Abstract 


Aims: Retrorectal tumors are rare, mostly benign tumors and named due to their localization. Diagnoses of these tumors are usually delayed because of nonspecific complaints and symptoms. Magnetic resonance imaging has beneficial uses both for diagnosis and treatment. In this study, we reviewed a case series of retrorectal tumors.
Subjects and Methods: The patients who were diagnosed with retrorectal tumors between 2008 and 2015 were analyzed. This investigation was conducted at a Tertiary Education and Research Hospital. Sixteen patients were included in this study. Patients' demographic data, imaging workups, surgical operation reports, pathologic examination results, postoperative complications, and follow-up results were examined. Descriptive statistics, median, and standard deviation for continuous variables were used. The primary outcomes measured were diagnostic conflict, knowledge, and preference for surgery.
Statistical Analysis Used: Definitive statistical methods (mean, standard deviation, median, frequency, and percentage) were used to evaluate the study data.
Results: One patient refused operation and one was in preoperative preparation period. Fourteen of sixteen patients were operated. Two (14.3%) of operated patients have malignant histopathological result (one gastrointestinal stromal tumor, one ganglioneuroblastoma). Rest of the operated patients' histopathological reports was as follows: Four schwannomas, three epidermoid cysts, two tailgut cyst, one dermoid cyst, one teratoma, and one angiomyolipoma. Eight patients were operated by posterior incision, five patients with transabdominal approach, and one patient with combined approach.
Conclusions: Retrorectal tumors are rare cases, and treatment of retrorectal tumors is surgery and should be operated in referenced hospitals to avoid diagnostic and therapeutic problems.

Keywords: Radiological assessment, retrorectal tumors, surgical assessment


How to cite this article:
Kilic A, Basak F, Su Dur MS, Sisik A, Kivanc AE. A clinical and surgical challenge: Retrorectal tumors. J Can Res Ther 2019;15:132-7

How to cite this URL:
Kilic A, Basak F, Su Dur MS, Sisik A, Kivanc AE. A clinical and surgical challenge: Retrorectal tumors. J Can Res Ther [serial online] 2019 [cited 2019 Oct 21];15:132-7. Available from: http://www.cancerjournal.net/text.asp?2019/15/1/132/183192




 > Introduction Top


Retrorectal tumors are a rare, heterogeneous tumor group. Retrorectal space is a potential space and known as presacral space as well.[1],[2],[3],[4] Its borders consist rectum's fascia propria anteriorly, presacral fascia posteriorly, rectum's lateral ligaments, ureters and iliac vessels laterally, and rectum's Waldeyer fascia superiorly.[3],[4],[5] Benign and malign tumors that originate from different embryological tissues are mostly benign cystic tumors and can develop in retrorectal space.[4]

Although there are a number of classifications for these tumors, the one designed by Uhlig and Johnson is the most common.[3],[4],[6],[7],[8],[9] Patients usually present with nonspecific complaints such as pelvic pain, constipation, and urogenital complaints. Nonspecific complaints usually delay the time of diagnosis.[10] Diagnosis becomes more convenient as imaging techniques improve. Magnetic resonance imaging (MRI) is the most useful imaging modality in both diagnosis and treatment of these tumors.

Treatment of retrorectal tumors is surgery, and detailed examination is necessary for a successful outcome.[1],[4],[10] Biopsy is not usually recommended, yet it is stated that biopsy may be an option on inoperable tumors with a high possibility of malignancy.[1],[11],[12] Various approaches are available in surgical treatment; however, lately laparoscopic and transanal endoscopic microsurgery started to take place in the literature.[1],[3],[4],[11],[12],[13],[14],[15],[16]

Majority of articles in the literature in retrorectal surgery consists of case presentations and a low number of case series.[9],[11] In this study, we aim to evaluate examination, treatment, and follow-up results of the patients with retrorectal mass.


 > Subjects and Methods Top


We planned an observational study using retrospective review of prospectively collected data. The Institutional Review Board approved the study, and the universal principles of the Helsinki Declaration were applied. After the approval, we examined the patients diagnosed with retrorectal mass between 2008 and 2015.

Patients' demographic data were recorded. Complaints and previous pelvic surgical history were examined. We also evaluated imaging workups, medical treatments, and surgical operation reports with detail. Pathology and additional therapy reports were collected. Postoperative complications and short-term outcome in postoperative 30 days were examined. Postoperative follow-up protocol was planned; each 3 months in the 1st year, MRI at the end of that year and every 6 months in the 2nd year. Defecation and micturition problems were questioned at follow-ups.

Statistical analysis

We used descriptive statistics, median, and standard deviation for continuous variables, and number and percentage for categorical data in data analysis.


 > Results Top


A total of 16 patients were diagnosed as retrorectal tumors. Ten patients (62.5%) were female, 6 (37.5%) were male and female:male ratio was 1.7. Mean age was 41.4 ± 18.7 and ranged between 16 and 68. Documentation of the patients' demographic data, imaging detail, size of mass in imaging, surgical approach, and histopathological diagnosis is shown in [Table 1].
Table 1: Case documentation

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Fourteen of sixteen patients were operated. One of the remaining two patients refused the operation, whereas other was in preoperative preparation period. Eight patients were operated by posterior approach, five patients with transabdominal approach, and one patient with combined approach. All patients were evaluated and operated by the same surgical team (AK, FB, AS, and AEK), who are experienced with retrorectal area owing to excessive colorectal surgery.

Two (14.3%) of the 14 operated was malign (one gastrointestinal stromal tumor [GIST], one ganglioneuroblastoma). Rest of the patients (n = 1 2, 85.7%) were benign (four schwannoma, three epidermoid cysts, two tailgut cyst, one dermoid cyst, one teratoma, and one angiomyolipoma). All patients had MRI preoperatively; five had computed tomography (CT) in addition. All radiological studies were examined by same experienced radiologist (shaded surface display). Mean mass size in MRIs was 8.3 cm, ranged between 2 and 19 cm.

Two of the patients (number 10 and 11) admitted with rectal bleeding and diagnosed with GIST via biopsy results from ulcerated hemorrhagic lesions. Patient number 10 received tyrosine kinase inhibitor, imatinib, preoperatively due to large mass located in pelvis [Figure 1]. After 3 months of neoadjuvant therapy, reduction in size of the mass was observed, and abdominoperineal resection was applied. Other GIST patient (number 11) received tyrosine kinase inhibitor, imatinib, in attempt to perform sphincter-preserving surgery. After treatment, she was planned for surgery; however, she rejected the operation.
Figure 1: Multiplanar magnetic resonance image views of patient number 10 with gastrointestinal stromal tumor. (a) Sagittal view showing mass between white arrows, (b) coronal view showing mass (white arrow), (c) axial view showing mass (white arrow)

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Relevant past medical history of patient number five revealed that she received nerve root block previously due to back pain. Patient number seven was operated on with diagnosis of pilonidal sinus in another hospital previously and admitted with infected presacral lesion in the 30th postoperative day. Radiologic examination showed a fistulized retrorectal cystic lesion and the mass was resected after antibiotherapy. Histopathological evaluation was reported as epidermoid cyst.

Patient number 14 was operated in another hospital previously and assessed as unresectable. Biopsy report was suspicious for liposarcoma. Patient admitted to our center with frequent urination and difficulty in defecation due to pressure to bladder and rectum. After new imaging, MRI [Figure 2] and CT [Figure 3] showing hypervascular retrorectal mass. He was operated with abdominal approach and during resection, ureter injury, and pelvic hemorrhage occurred. The ureter was repaired with stent. Pelvic packing was applied for the hemorrhaging presacral veins. Reoperation was performed for depacking and vascular repairment after 48 h when hemodynamic stabilization obtained in Intensive Care Unit. The patient had no complications postoperatively, and histopathological report was angiomyolipoma.
Figure 2: Multiplanar magnetic resonance image views of patient number 14 with angiomyolipoma show the mass (white arrow). (a) Sagittal view, (b) coronal view, (c) axial view

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Figure 3: Multiplanar contrast-enhanced computed tomography views of patient number 14 with angiomyolipoma. (a) Sagittal view showing mass (white arrow), (b) coronal angiography view showing hypervascularization between white arrows, (c) axial view showing hypervascular mass between white arrows

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Four patients (number 1–4), two women and two men, operated for schwannoma. Primary complaints were back pain. Pelvic mass was noticed during appendectomy operation in one of the patients (number 1). Biopsy was performed and reported as neuroepithelial tumor. The mass was resected with secondary definitive surgery. Three of four patients had sacral bone destructions in variable proportions. All four patients were operated with abdominal approach.

Patient number 12 had multicystic infra- and supra-levator cysts with different sizes that adhered to rectal wall. Cysts' were difficult to dissect, and some cysts ruptured during dissections. Pathology reported dermoid cyst.

Patient number 13 had CT reports consistent with teratoma containing heterogeneous cysts with bone structures, and histopathological evaluation reported teratoma [Figure 4].
Figure 4: Multiplanar computed tomography views of patient number 13 with teratoma. (a) Axial view showing cystic lesion containing bone structures, (b) sagittal view of the lesion

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The average follow-up period was 937 ± 738 days (range 102–2252) for all patients. No mortality or severe postoperative complications were observed in our series. Temporary defecation and micturition problems were detected in two of the operated patients. Bone metastasis was detected at follow-up in patient (number 15) with ganglioneuroblastoma, and patient received radiotherapy. No other metastasis or recurrence was observed.


 > Discussion Top


Retrorectal tumors are rare cases and surgeons studying other than referenced hospitals encounter these cases once or twice in their careers.[4] Rare cases such as these can be a problem for the surgeons with insufficient experience. We understand that this situation is present because most of the publications for this subject are mostly case reports. Inadequate surgery can be performed to a certain number of patients even in referenced hospitals.[12] In our series, two patients can be evaluated as inadequate intervention before the admission to our hospital. One of them was referred after the patient was operated with diagnosis of pilonidal sinus in another hospital. Other patient was determined as unresectable in the first operation. We resected the mass with acceptable complications.

Nonspecific pelvic and rectal complaints are in the foreground while most of the patients are asymptomatic. However, these complaints may delay diagnosis.[1],[17] In the study of Macafee et al.,[11] complaints of the patients were mostly pelvic, sacral, lower back and perianal pain, discomfort, changes in bowel habits, difficulty in defecation, and tenesmus. One patient's tumor diagnosed during cesarean section. As the mass grows, the pressure on organs rises and different complaints and symptoms occur. Difficulty in defecation, pelvic pain, and tenesmus came in the foreground for the lesions compressing the rectum. In our study, patients applied to the hospital mostly with nonspecific, back, pelvic pain and constipation. One of the patients (number 5) received nerve root block before admission to our clinic. Patient's pain was possibly associated with the mass. MRI omitted the mass, and the patient received inappropriate treatment. The patient with angiomyolipoma had urinary complaints such as frequent urination and feeling of incomplete bladder emptying in the foreground due to pressure to the bladder. Two patients with rectal GIST were diagnosed after examinations due to rectal bleeding.

Benign tumors are usually detected incidentally during gynecological or other examinations for other reasons. However, pelvic pain is the most distinct complaint in malign retrorectal tumors.[2],[12] This may be the answer of why benign lesions are detected mostly in female. In our study, ten patients (62.5%) were female, and female:male ratio was 1.7. Nine of ten (90%) female patients were benign and one of them was malign. However, this patient was diagnosed with biopsy and refused the operation. Four of six (66.7%) male patients were benign, and one was malign. Remaining patient was in preoperative period and not diagnosed histopathologically yet.

Rectal examination is highly important, and palpated mass ratio is 97% during digital rectal examination in the publication of mayo clinic experience of Jao et al.[18] Rectal examination is not only important for the diagnosis but also for the evaluation of the upper verge of the mass and to determine the method of surgery.[4],[18]

Diagnosis becomes easier as the imaging methods, especially CT and MRI becomes more convenient. However, misdiagnosis is usually the result of insufficient examination and inexperience. MRI gives hints for distinction between malign and benign masses. Solid, irregular-shaped, infiltrative, heterogeneous formed lesions and patterns of contrast are the signs of malignancy. Cystic, smooth bordered, noninvasive lesions without contrast are mostly benign. Invasion and destruction of bone structures can be detected, and this can prevent a complicated and time-consuming surgery.[11] In our series, most of the patients were diagnosed radiologically consistent with pathological evaluation. Therefore, it can be said that examination of an experienced radiologist is required for preoperative differential diagnosis.

Although retrorectal tumors that are a heterogeneous group are mostly benign, surgery is suggested to resolve the symptoms and prevent possible complications and transformation to malignancy. The malignant transformation of cystic lesions is mentioned as rare in literature.[1],[4],[15] All the cystic cases were benign in our study.

There is a consensus that primary treatment of retrorectal tumors should be surgery, biopsy during diagnosis phase is contradictive beside exceptions. Although “biopsy is unnecessary” view is popular, biopsy may be performed for tumors with high risk of malignancy and that are hard to remove or not resectable and convenient for neoadjuvant therapy. We should keep in mind that while carrying out a biopsy for malignant tumors, there is a chance of seeding to the biopsy tract. Transrectal and transvaginal biopsy should not be performed.[4],[8],[12] In our study, biopsy was performed during rectosigmoidoscopy in two patients who had rectal bleeding. Both of these patients were reported as GIST. One patient was biopsied during laparotomy, reported as suspicious in terms of liposarcoma in another hospital and report after definitive resection came back as angiomyolipoma. One patient had undergone appendectomy, a mass was seen during exploration and the biopsy report came back as benign neuroepithelial tumor. Resected mass was reported as schwannoma.

There are a number of classifications for retrorectal tumors.[3],[4],[7],[8],[9] Mostly used one is from Uhlig and Johnson, and tumors were split up into five chapters; congenital, inflammatory, neurogenic, osseous, and others.[6] Congenital tumors make up 55–70% of the tumors from this group and contain developmental cysts, chordomas, anterior meningocele, rectal duplication cysts, and adrenal rest tumors.[4] In our study, 7 (46.6%) of 15 diagnosed patients (epidermoid, dermoid cyst, teratoma, and tailgut cyst) were congenital, 5 (33.3%) (schwannoma and ganglioneuroblastoma) were neurogenic, and 3 (20%) (GIST and angiomyolipoma) were in others category. Three (20%) of these 15 patients were malignant and others were benign. Malignant tumors are more frequently present in men and seen in elder years than benign tumors.[4] Two of the malignant tumors in our series were GIST, and both patients were in their seventh decade, the other one is ganglioneuroblastoma. While ganglioneuroblastoma is stated as an intermediate tumor in literature, our patients had developed metastases during follow-ups. However, chordomas are the most seen malignant retorectal tumors in literature, there was no chordoma in our series.[4]

Pappalardo et al.'s[7] classification has three subgroups for retrorectal tumors; first group, originating from retrorectal space; second, originating from sacrum or spinal cord and developing anteriorly; and third, originating from rectum and developing posteriorly. It has been reported that first and third group can usually be operated by general or colorectal surgeons. Orthopedic surgeon or neurosurgeon support is needed for the second group. In our series, tumors with sacrum invasion were operated as a team of general, gynecologic, and/or neurosurgeons. It is indicated that for tumors above third–fourth sacral vertebra abdominal or combined approach is more convenient. It was also indicated that no pelvic stabilization is needed for third sacral vertebra or below, but stabilization must be maintained for above.[4],[11] Posterior approach (Kraske procedure) may be more convenient for tumors below third sacral vertebra. Including coccyx to the resection depends on its connection to the tumor and acquiring enough operation site. There is no need to resect coccyx routinely. Tumor-rectum connection should be regarded in both posterior and anterior approach. Rectum injury should be avoided and be repaired immediately. Injury should be checked intermittently by rectal examination and rectum borders examined in posterior approach.[1],[17],[19] In our study, we have also encountered rectum injury in a patient with tailgut cyst connected with rectum wall and it was repaired primarily. Retrorectal developmental cysts are highly adherent to coccyx. Their wall structure is thin, and they inevitably perforate during dissection from coccyx.[15] Controlled aspiration of big cysts may make it more convenient to operate.

There has been published case series and reports on minimally invasive, laparoscopic, and endoscopic surgical techniques of retrorectal tumors, whereas posterior, abdominal, and combined approaches have been accepted in the literature before. Main limitation for this study was that we presented a limited number of cases with heterogeneous group of retrorectal tumors. Therefore, we could not associate any diagnostic and prognostic factors with these tumors. We presented case series of retrorectal tumors with an average follow-up of 2.6 years. Although this follow-up seems sufficient, future studies with larger number of cases are required to evaluate this association.


 > Conclusions Top


Retrorectal masses are a rare heterogeneous tumor group, mostly with nonspecific symptoms, and there may be difficulties in diagnosis and treatment for some cases that require experience and multidisciplinary approach.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

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Chéreau N, Lefevre JH, Meurette G, Mourra N, Shields C, Parc Y, et al. Surgical resection of retrorectal tumours in adults: Long-term results in 47 patients. Colorectal Dis 2013;15:e476-82.  Back to cited text no. 1
    
2.
Dozois E, Jacofsky D, Dozois R. Presacral tumors. The ASCRS Textbook of Colon and Rectal Surgery. In: Wolff BG, Fleshman JW, Beck DE, Pemberton JH, Wexner SD, Church JM, et al, editors. Switzerland: Springer International Publishing AG; 2007. p. 501-14.  Back to cited text no. 2
    
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Mentes BB, Kurukahvecioglu O, Ege B, Karamercan A, Leventoglu S, Yazicioglu O, et al. Retrorectal tumors: A case series. Turk J Gastroenterol 2008;19:40-4.  Back to cited text no. 3
    
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Hobson KG, Ghaemmaghami V, Roe JP, Goodnight JE, Khatri VP. Tumors of the retrorectal space. Dis Colon Rectum 2005;48:1964-74.  Back to cited text no. 4
    
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Sagar AJ, Tan WS, Codd R, Fong SS, Sagar PM. Surgical strategies in the management of recurrent retrorectal tumours. Tech Coloproctol 2014;18:1023-7.  Back to cited text no. 5
    
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Uhlig BE, Johnson RL. Presacral tumors and cysts in adults. Dis Colon Rectum 1975;18:581-9.  Back to cited text no. 6
    
7.
Pappalardo G, Frattaroli FM, Casciani E, Moles N, Mascagni D, Spoletini D, et al. Retrorectal tumors: The choice of surgical approach based on a new classification. Am Surg 2009;75:240-8.  Back to cited text no. 7
    
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Messick CA, Hull T, Rosselli G, Kiran RP. Lesions originating within the retrorectal space: A diverse group requiring individualized evaluation and surgery. J Gastrointest Surg 2013;17:2143-52.  Back to cited text no. 8
    
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Kesici U, Sakman G, Mataraci E. Retrorectal/presacral epidermoid cyst: Report of a case. Eurasian J Med 2013;45:207-10.  Back to cited text no. 9
    
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Sagar AJ, Koshy A, Hyland R, Rotimi O, Sagar PM. Preoperative assessment of retrorectal tumours. Br J Surg 2014;101:573-7.  Back to cited text no. 10
    
11.
Macafee DA, Sagar PM, El-Khoury T, Moles N, Mascagni D, Spoletini D, et al. Retrorectal tumours: Optimization of surgical approach and outcome. Colorectal Dis 2012;14:1411-7.  Back to cited text no. 11
    
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Glasgow SC, Birnbaum EH, Lowney JK, Fleshman JW, Kodner IJ, Mutch DG, et al. Retrorectal tumors: A diagnostic and therapeutic challenge. Dis Colon Rectum 2005;48:1581-7.  Back to cited text no. 12
    
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Duclos J, Maggiori L, Zappa M, Ferron M, Panis Y. Laparoscopic resection of retrorectal tumors: A feasibility study in 12 consecutive patients. Surg Endosc 2014;28:1223-9.  Back to cited text no. 13
    
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Fong SS, Codd R, Sagar PM. Laparoscopic excision of retrorectal tumours. Colorectal Dis 2014;16:O400-3.  Back to cited text no. 14
    
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Zhou JL, Wu B, Xiao Y, Lin GL, Wang WZ, Zhang GN, et al. Alaparoscopic approach to benign retrorectal tumors. Tech Coloproctol 2014;18:825-33.  Back to cited text no. 15
    
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Serra Aracil X, Gómez Díaz C, Bombardó Junca J, Mora López L, Alcántara Moral M, Ayguavives Garnica I, et al. Surgical excision of retrorectal tumour using transanal endoscopic microsurgery. Colorectal Dis 2010;12:594-5.  Back to cited text no. 16
    
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Buchs N, Taylor S, Roche B. The posterior approach for low retrorectal tumors in adults. Int J Colorectal Dis 2007;22:381-5.  Back to cited text no. 17
    
18.
Jao SW, Beart RW Jr., Spencer RJ, Reiman HM, Ilstrup DM. Retrorectal tumors. Mayo clinic experience, 1960-1979. Dis Colon Rectum 1985;28:644-52.  Back to cited text no. 18
    
19.
Troja A, El-Sourani N, Antolovic D, Raab HR. Transsphincteric tumor resection in case of a pararectal solitary fibrous tumor. Int J Surg Case Rep 2016;19:159-62.  Back to cited text no. 19
    


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