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© Borgis - Nowa Medycyna 2/2017, s. 49-53
*Szymon Głowacki1, Zbigniew Suwała1, Karol Łukasiak1, Tomasz Pokładowski1, Michał Kwiatkowski2
Retrorectal tumour – a case report
Guz przestrzeni zaodbytniczej – opis przypadku
1Department of Surgery, Independent Public Complex of Health Care Facilities, Sierpc
Head of Department: Szymon Głowacki, MD, PhD
2Department of Pathomorphology, Provincial Hospital in Płock
Head of Department: Michał Kwiatkowski, MD
Streszczenie
Autorzy prezentują przypadek pacjentki operowanej z powodu zmiany w przestrzeni przedkrzyżowej/zaodbytniczej o charakterze torbieli epidermalnej. Pierwotnie wywodzi się ona z torbieli wrodzonych i należy do zmian łagodnych. Zmiana została zdiagnozowana przypadkowo w trakcie badania per rectum. Głównymi dolegliwościami były niespecyficzne bóle w podbrzuszu oraz przewlekłe zaparcia. Chora zgłaszała również nasilenie objawów związanych z chorobą zwyrodnieniową kręgosłupa. Zaplanowano i przeprowadzono zaawansowaną diagnostykę tej okolicy: ultrasonografię transrektalną oraz tomografię komputerową jamy brzusznej i miednicy z kontrastem. Oba badania potwierdziły patologiczny obszar o charakterze torbieli. Pozwoliło to na eliminację chorób typowych dla tej okolicy, takich jak ropień okołoodbytowy, przetoka czy szczelina odbytu. W kolonoskopii wykluczono infiltrację ściany odbytnicy. Nie wykonywano biopsji przedoperacyjnej. Zakwalifikowano chorą do operacji klasycznej z dojścia dolnego. Zmianę wycięto w całości. Przebieg pooperacyjny bez powikłań. Kontrola po 3 miesiącach nie wykazała wznowy. Zmiany okolicy zaodbytniczej, przedkrzyżowej są zmianami trudnymi diagnostycznie, rozpoznawanymi późno, często przebiegają bezobjawowo. Prawidłowa diagnostyka i właściwa technika operacyjna pozwalają na całkowite wyleczenie.
Summary
We present a case of a female patient operated on due to a lesion in the form of an epidermoid cyst located in the retrorectal/presacral region. The lesion originates from congenital cysts and has a benign nature. The lesion was discovered accidentally during a routine rectal exam. The patient’s main prediagnostic complaints included nonspecific pain in the lower abdominal and lesser pelvic region, and chronic constipation. The patient also complained of pain exacerbation due to a degenerative spinal disease. An extensive diagnostic approach including transrectal ultrasound and contrast enhanced computed tomography was planned and implemented. Both of these techniques confirmed the pathological cystic nature of the lesion and allowed for exclusion of other lesions typical of this area, such as perianal abscess, anal fissure or fistula in ano. Colonoscopy revealed no mucosal involvement, no preoperative biopsy was obtained. The patient was scheduled for a surgery via a perianal incision, which allowed for a complete excision. The postoperative period was uneventful. A 3-month follow-up exam showed no recurrence. We wish to point out that retrorectal/presacral lesions comprise a diagnostic challenge as they are often clinically asymptomatic and thus discovered late. A correct diagnosis followed by a proper surgical technique allows for a complete curative effect.
Słowa kluczowe: leczenie operacyjne.
Introduction
Retrorectal-presacral tumours most often present as epidermoid cysts (1). They are usually congenital and often asymptomatic due to their location. The symptoms are nonspecific and often confused with many other diseases of this region, such as discopathy, peritoneal adhesions after gynaecological procedures, proctological conditions of the anus, such as fissures, fistulas and haemorrhoids. The clinical course may be violent. This is associated with the infection of the cyst, accidental injury and damaged cystic wall leading to local complications. The diagnosis and surgical treatment are a difficult challenge for the doctor.
Case report
We present a case of a 60-year-old female patient with non-specific lesser pelvic symptoms accompanied by periodical constipations. Medical history of lumbosacral discopathy. The symptoms of discopathy increased after a lumbosacral injury the year before.
Physical examination: the patient was in good overall condition. No thoracic or abdominal abnormalities.
Local condition: rectal examination in Sims position – patient positioned on the left side with knees drawn up to abdomen; a palpable pathological mass about 3 cm in size above the anal canal, on the posterior wall. A soft lesion about 5-6 cm in diameter, located behind and posterior to the rectum.
Additional exams
Colonoscopy: colonoscopy confirmed rectal wall deformation with no signs of mucosal involvement.
Computed tomography: the CT revealed a 65 x 55 mm cystic lesion located in the posterior rectum, with no signs of invasion.
Transrectal ultrasound: transrectal ultrasound revealed an extensive cystic lesion. The lesion did not infiltrate the rectal wall and was strongly fixed to the sacrum. It posteriorly involved the rectum and extended on the right side. The imaging showed no tumour infiltration. The grade of continence was measured during proctological examination and based on the Wexner score system. The patient was scheduled for surgical treatment. A total resection of the lesion was performed (fig. 1). The postoperative period was uneventful; the patient was discharged home in good condition and with no signs of incontinence, with instructions for a further ambulatory follow-up.
Fig. 1. A cross-section of the resected cyst
Histopathological findings
Microscopic histopathological examination revealed a retrorectal epidermoid cyst. Further interpretation: a benign lesion, a cyst lined by keratinising stratified squamous epithelium, with no skin appendages. The presence of resorptive granulation tissue in the cystic lumen, wall and adjacent tissues indicates that the cyst probably perforated in the past.
Discussion
Retrorectal-presacral lesions are rare with the incidence of 1:40,000 patients, and are more common in middle-aged females (ratio 3:1) (1). Their nature is described using different classification systems. We used the classification system developed by Uhlig and Johnson (2). According to this classification, congenital, neurogenic, osseous, inflammatory and mixed cysts may be distinguished. The system also divides between benign and malignant cysts (3). Dahan et al. distinguished congenital, epidermoid, dermoid and intestinal cysts, with the latter differentiated into cystic hamartomas and rectal duplication cysts (4). The first division is based on the type of epithelium lining the lesion (5, 6). Appropriate diagnosis requires clinical experience. These lesions are difficult to diagnose. Palpation is the basic proctological method. However, it is not possible to determine the nature of the lesion or plan surgical technique without imaging. Computed tomography, magnetic resonance and transrectal ultrasonography are recommended tools to evaluate the above mentioned characteristics (7-9). These techniques allow to assess the topography of cysts relative to adjacent organs, vessels and nerves as well as to obtain data on potential features indicating malignancy. This is primarily indicated by rectal wall or sacral involvement, the density of the intracystic fluid and wall thickness. Adenocarcinomas, neuroendocrine or squamous cell carcinoma have been reported to occur in this region in the available literature (10-12). The possible occurrence of these tumours confirms the role of imaging techniques. In the presented case, abdominal and pelvic CT as well as transrectal ultrasound were performed and allowed for a preoperative diagnosis of a benign lesion. The patient was operated on from the posterior approach. Posterior approach is recommended in cysts located distally to S3, whereas anterior approach is used in cysts located proximally to S3 level (13-15). Preoperative biopsy is also debatable. Although some authors recommend biopsy, a number of reports confirm that a total resection of the lesion is an appropriate management strategy (16). At this point, it is worth paying attention to three most important complications that may be encountered in this disease. These include cyst infection, bleeding and the risk of cancer spread, e.g. squamous cell carcinoma or adenocarcinoma in the case of intestinal cysts. Therefore, total resection is the treatment of choice (17-19). The differentiation should also include rare genetic conditions typical of this region. We also wish to emphasise the importance of the Currarino syndrome with the triad of presacral symptoms in the diagnostic process (20). The multitude of pathologies in this region and their non-specificity confirm the complexity of this issue.
In our case, a total resection followed by histopathological examination was implemented. A benign lesion was confirmed. A follow-up after 3 months showed no local recurrence. The patient reported no signs of incontinence and the tension of the sphincter muscle, which was evaluated during rectal examination, was considered normal. Continence status was evaluated based on Wexner score (21).
Conclusions
1. Retrorectal-presacral lesions are diagnostically difficult, often asymptomatic and diagnosed at late stages.
2. Proper diagnosis and appropriate surgical technique allow for a complete curative effect in most cases.
Piśmiennictwo
1. Kołodziejczak M, Warzecha W, Sudoł-Szopińska I, Stusińska M: Atypically located epidermoid cyst as a cause of proctalgia – a case report. Nowa Med 2015; 4: 109-113.
2. Uhlig BE, Johnson RL: Presacral tumors and cysts in adults. Dis Colon Rectum 1975; 18(7): 581-589.
3. Dozois EJ, Jacofsky DJ, Dozois RR: Presacral tumors. [In:] Wolff BG, Fleshman JW, Beck DE et al. (eds.): The ASCRS Textbook of Colon and Rectal Surgery. Springer, New York 2007: 501-514.
4. Dahan H, Arrivè L, Wendum D et al.: Retrorectal developmental cysts in adults: clinical and radiologic-histopathologic review, differential diagnosis, and treatment. Radiographics 2001; 21(3): 575-584.
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8. Johnson AR, Ros PR, Hjermstad BM: Tailgut cyst: diagnosis with CT and sonography. AJR Am J Roentgenol 1986; 147(6): 1309-1311.
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16. Merchea A, Larson DW, Hubner M et al.: The value of preoperative biopsy in the management of solid presacral tumors. Dis Colon Rectum 2013; 56: 756-760.
17. 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(12): 2143-2152.
18. Gong L, Liu W, Li P, Huang X: Transsacrococcygeal approach for resection of retrorectal tumors. Am Surg 2015; 81(6): 569-572.
19. Chèreau N, Lefevre JH, Meurette G et al.: Surgical resection of retrorectal tumours in adults: long-term results in 47 patients. Colorectal Dis 2013; 15(8): e476-482.
20. Cartanese C, Franco F, Guastadisegno C et al.: Retrorectal multilocular cyst in a adult female. Case report and review of literature. Ann Ital Chir 2015; 86(ePub).
21. Jorge JMN, Wexner SD: Etiology and management of fecal incontinence. Dis Colon Rectum 1993; 36: 77-97.
otrzymano: 2017-04-12
zaakceptowano do druku: 2017-05-04

Adres do korespondencji:
*Szymon Głowacki
Oddział Chirurgiczny Samodzielny Publiczny Zespół Zakładów Zdrowotnych
ul. Słowackiego 32, 09-200 Sierpc
tel. +48 (24) 275-85-16
szymon.glowacki@onet.pl

Nowa Medycyna 2/2017
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