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REVIEW ARTICLE
Year : 2013  |  Volume : 9  |  Issue : 3  |  Page : 343-347

Sacrococcygeal neoplastic lesions


1 Department of General Surgery, University Hospital of Ioannina, Ioannina, Greece
2 Department of Neurosurgery, University Hospital of Ioannina, Ioannina, Greece

Date of Web Publication8-Oct-2013

Correspondence Address:
Georgios Lianos
Department of General Surgery, University Hospital of Ioannina, Ioannina, Greece, St. Niarchou Avenue 45500, 5 Platonos Avenue 45221 Katsikas Ioannina
Greece
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.119297

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 > Abstract 

Sacrococcygeal neoplastic lesions are very rare entities and therefore, a diagnostic and therapeutic challenge. They include developmental cysts, inflammatory, neurogenic, osseous and miscellaneous lesions. In this review, we discuss the incidence, symptoms, characteristics, diagnostic methods and treatment strategies of these enigmatic lesions. We have to highlight the high index of suspicion that is necessary when dealing with chronic sacrococcygeal pain or multiple perianal abscesses or persistent perianal drainage, because any misinterpretation may result in unfavorable outcomes.

Keywords: Diagnosis, incidence, sacrococcygeal lesions, treatment


How to cite this article:
Lianos G, Alexiou G, Zigouris A, Voulgaris S. Sacrococcygeal neoplastic lesions. J Can Res Ther 2013;9:343-7

How to cite this URL:
Lianos G, Alexiou G, Zigouris A, Voulgaris S. Sacrococcygeal neoplastic lesions. J Can Res Ther [serial online] 2013 [cited 2019 Nov 21];9:343-7. Available from: http://www.cancerjournal.net/text.asp?2013/9/3/343/119297


 > Introduction Top


Neoplastic lesions of the sacrococcygeal region are very uncommon and are rarely encountered in current surgical practice. Despite the use of new imaging modalities, they remain a diagnostic and therapeutic challenge. [1],[2] They range from benign cysts to malignant masses invading pelvic structures and may be congenital or acquired. [3] The true incidence is difficult to assess but is reported as one case out of 40.000 admissions to large referral centers. Interestingly, it is reported that the majority of general surgeons during their career practice treat only one case of sacrococcygeal tumor. [4],[5] The first successful resection of a sacrococcygeal tumor was made with combined anteroposterior approach in 1945 by Bowers after an initial posterior unsuccessful approach for a presumed chordoma, because of extensive vascular collateralization. [6]

The sacrococcygeal region, also referred as "presacral region" or "retrorectal space," is an anatomic zone occupied by connective tissue, blood, nerves and lymphatic vessels. This space is defined anteriorly by the fascia propria of the rectum and posteriorly by the presacral fascia overlying the sacrum and coccyx. Laterally is bordered by the lateral stalks of the rectum, the iliac vessels and the ureters. [7] Embryologic structures, neural elements, bone and many types of tissue in this region may lead to the development of different lesions. The sacrococcygeal lesions are categorized into five categories (1) congenital (2) inflammatory (3) neurogenic (4) osseous and (5) miscellaneous. Another classification divided them into benign and malignant.[Table 1]. [8]
Table 1: Classification system

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 > Developmental Cysts Top


The developmental cysts represent a 60-70% of all congenital sacrococcygeal lesions. They can originate from any of the three germ layers. The female to male ratio is 5:1. These lesions are often misdiagnosed as a perirectal abscess and they can be divided into the following groups: epidermoid cysts, dermoid cysts, enterogenous cysts, tail gut cysts and teratomas. [9],[10] The correct diagnosis of these lesions is very difficult, leading to repeated drainage procedures prior to achieve the final diagnosis. [11] Although developmental cysts are often asymptomatic, some patients may appear with symptoms resulting from the local mass effect, as constipation, lower abdominal pain, urinary disturbances. There is often a palpable retrorectal mass at the rectal examination. Sometimes these cysts are presented with a complication such as infection, hemorrhage and rarely with malignant degeneration. [12]

Epidermoid and dermoid cyst

The epidermoid cysts are benign unilocular lesions that are composed of stratified squamous epithelium. Dermoid cysts are not composed only of stratified squamous cells, but they can have skin appendages and organelles (hair follicles, sebaceous cysts etc.). [12] Epidermoid and dermoid cysts most commonly appear in females, typically benign and although they do not communicate with the rectum, around a 30% of them seems to be infectious.

Cystic rectal duplication

Cystic rectal duplication is a rare entity, representing a 5% of all developmental cysts. Cystic rectal duplication arises from the hindgut and has endodermal origin. It is reported that these lesions have a dominant cyst, associated with several smaller cysts, called 'daughter' cysts or 'satellite' cysts. There are three histologic criteria defining cystic rectal duplication: (1) continuity with the rectum (2) a smooth-muscle coat in two layers and (3) a mucosal lining, which is often similar to rectal mucosa. It sometimes contains zones of ectopic tissue such as gastric mucosa, pancreatic tissue or urothelium. Although these lesions are benign, cases of malignant degeneration have been reported. [13],[14],[15]

Amartoma

Cystic amartomas, also called tailgut cysts or mucus-secreting cysts, are more common in women and are benign in the majority of cases. They are often lined with transitional epithelium and are reported to derive from the hindgut. These lesions may be multicystic and filled with mucoid contents and although benign, malignant degeneration has been reported. Adenocarcinoma is the most frequent histological finding in these cases. [16]

Teratoma

The prevalence of sacrococcygeal teratoma is one out of 40.000 live births with a significant female predilection 4-1. The most common site of origin is believed to be the sacroccocygeal area, followed by ovaries, neck, retroperitoneum, mediastinum, testicles and central nervous system. In the modern era the largest series of sacrococcygeal teratomas was analyzed by Gross et al. in 1951. [17],[18] These masses are lesions arising from totipotential cells. They have tissues derived of all three germ layers (ectoderm, mesoderm, and endoderm). Their origin is uncertain, but is believed to arise early in gestation from the totipotential cells of Hensen's node, which is a remnant of the primitive streak in the coccygeal region. [19],[20] Teratomas are classified as benign (mature) and malignant or immature (composed of embryogenic elements). Many authors report that approximately 18% of patients with sacrococcygeal teratoma have also other congenital anomalies (sacral agenesis, spinal dysraphism, meningoceles, cardiac or gastrointestinal disturbances). [21] The infantile teratomas are classified into four types: (1) Predominantly external with minimal presacral component (2) Present externally but with significant intrapelvic extension (3) Apparent externally but predominantly a pelvic mass extending into the abdomen (4) Presacral with no external presentation. The possibility of malignant degeneration is greater during neonatal period and early childhood. Many authors support that the coccyx is rarely involved unless the tumor is malignant. [22] The frequence of malignancy in sacrococcygeal teratomas ranges from 10% to 50%. It is believed that a 7% of masses existing at birth, if not treated, will become malignant after 4 months, while malignant transformation in adult teratomas is estimated at about 30%. [23] The sacrococcygeal teratomas in adults are extremely rare. While teratomas in infants are externally visible at the 80-90% of cases, in adults are mostly confined to the pelvis. The diagnosis is very difficult and the lesion may be present as a fistula in anal region before diagnosis is established. In some cases, rupture of teratoma in the rectum has been described. [24],[25],[26] Despite the fact that only a 4% of teratomas at birth are malignant, those found later tend to be more aggressive and are associated with a poor outcome. Interestingly, a 30% of adult teratomas harbor malignancy at the time of resection. Computed tomography (CT) and magnetic resonance imaging (MRI) are the diagnostic 'gold standard' methods for these neoplasms. The surgical wide and gross total resection is the only therapeutic option, with routine attempt to remove also the coccyx, as it may contain the nidus of totipotential cells. In cases where coccyx was not removed, high rates of recurrence have been reported. [27],[28]


 > Inflammatory Top


Sacrococcygeal inflammatory lesions arise from infection source: perineal abscess or pelvic abscess in Crohn's disease, diverticulitis or other intraabdominal infection. Many times malignant lesions of the sacrococcygeal area are misdiagnosed as inflammatory masses, resulting in unfavorable outcomes. [29]


 > Neurogenic Top


Neurofibroma

Neurofibroma is exceedingly rare and only 17 cases have been reported to date. [30] Paul et al. recently reported a case of a 38-year-old woman with chronic pelvic pain that was caused by a neurofibroma. The authors managed to totally excise the tumor laparoscopically. [30]

Ependymoma

Ependymomas outside the central nervous system are exceedingly rare. Recently, Alexiou et al. [31] reported a case of a 13-years-old girl with a sacrococcygeal tumor misdiagnosed as a pilonidal sinus. These tumors usually have a good prognosis if complete excision is performed. [32] The role of radiotherapy is dubious, nevertheless, long term follow-up is mandatory.

Neurilemoma (Schwannoma)

Schwannoma, originating from Schwann cells, is a tumor of peripheral nerves. This tumor type usually affects the intracranial acoustic nerve and spinal nerves. Presacral schwannomas are very rare and only few cases have been reported. [33]

Ganglioneuroma

Ganglioneuromas are benign slow-growing masses and usually do not recur after complete surgical extirpation. These tumors are composed of ganglion cells and Schwann cells. [34] To date only 17 cases have been reported in the literature. [35]


 > Osseous Top


Osteoma

Osteoid osteoma is a benign osteoblastic tumor, usually affecting long bones. About 7-20% of osteoid osteoma involves the spine. Nevertheless, sacrococcygeal tumors are very rare. This tumor usually presents in young patients and there is a male predilection. [34]

Osteogenic sarcoma

Osteogenic sarcoma is the second most common primary bone tumor and usually affects children. An osteochodroma may dedifferentiate to a sarcoma. An X-ray usually shows both lytic and osteoblastic changes. [35] Only few cases have been reported in the sacrum, whereas metastases in the spine is the most common.

Rare tumors

In the sacrum other rare osseous tumors involve Ewing's sarcoma, chondromyxosarcoma and giant cell tumor.


 > Miscellaneous Top


Miscellaneous' category (15-20%) includes benign masses (lipoma, fibroma, leiomyoma, hemangioma, lymphangioma), malignant lesions (liposarcoma, fibrosarcoma, leiomyosarcoma), lymphomatous masses and metastases (usually from the rectum).

Lipoma

Lipomas are found into subcutaneous space and are composed of fatty tissue; they may be soft, single or multiple lobulated and increase with time. Sacrococcygeal lipomas are rare and benign, but they must be recected because of the low possibility of malignant degeneration; when they achieve a diameter greater than 8 cm they should be investigated for malignancy. [36]

Fibroma

Fibromas are extremely rare and have a high incidence of local recurrence. In the pediatric population it has been described a rare entity called fibromatosis, presented as a sacrococcygeal mass, which is a musculoaponeurotic border line tumor that must be surgically resected. [37]

Angioma

Hemangiomas are presented as large extended lesions associated with venous, capillary or arteriovenous malformations. Cystic lymphangiomas (are presented) appear more frequently in childhood. The most common site is cervical and rarely sacrococcygeal. They appear as a well-defined, multicystic tumor and they may cause as complications hemorrhage and infection. [38]

Liposarcoma

Liposarcoma is a malignant mesenchymal neoplasm usually found in the upper thigh. In the sacrococcygeal region rarely arises from intermuscular fascia. It may be well or poorly differentiated and complete surgical resection is the definitive treatment. [39] The congenital fibrosarcoma is a rare pediatric soft-tissue tumor, that occurs usually in the first year of life and has a better prognosis compared to other more aggressive spindle cell sarcomas presented in pediatric age. According to differential diagnosis, it must be concerned that fibrosarcoma may simulate hemangioma because of its highly vascularization. [40] Most common sites are limbs, trunk, head and neck. The sacrococcygeal location is extremely rare and is reported only one case in literature of congenital infantile fibrosarcoma masqueranding a teratoma. [41]

Leiomyosarcoma

Leiomyosarcomas are originated from smooth muscle, main structural component of organs and vessels. These tumors can occur almost anywhere in the body. This type of tumor is commonly observed in uterus and in the gastrointestinal tract. Sacrococcygeal leiomyosarcomas are of rectal origin. Rectal leiomyosarcomas (malignant rectal stromal tumors) are rare (0.1%). They have a male predominance and originate from the hematogenous and local spread. The treatment is surgical with poor prognosis. [42],[43]

Lymphoma

Lymphomas are divided in true lymphomas (non-Hodgkin, Hodgkin) and post transplantation lymphoproliferative disorders. It is reported that among non-Hodgkin lymphoma, lymphoma Burkitt, Burkitt-like and B-cell lymphomas are more common and presented as abdominal or sacrococcygeal masses with involvement of iliac nodes. [44]

Carcinoid

Carcinoid tumors present as direct extension or metastatic from rectal carcinoid. 5-hydroxyindolacetic acid could be used as tumor marker and as a biochemical marker of recurrence as for carcinoid tumors in other location. [45],[46],[47] They should be treated surgically.


 > Diagnosis Top


The patient with sacrococcygeal neoplastic lesion is often asymptomatic. He may observe changes in bowel habits, constipation, diarrhea, frequent urge to defecate, as a result of tumor's compression to the rectum. [29],[48] Other symptoms include low back pain, sacral and perineal pain, urinary incontinence and hemorrhage. Early diagnosis is very difficult when the lesion has a small size. Diagnosis is made when the tumor is so large to cause symptoms by compression of adjacent structures. Digital rectal examination is essential for diagnosis of these lesions. In the majority of patients (80-90%) is detected a palpable mass in the digital rectal examination or is described a feeling of an extrarectal mass displacing the rectum anteriorly. [49],[50] Clinical diagnosis requires a high index of suspicion when dealing with patients with a medical history of perianal persistent drainage or multiple perianal abscesses or chronic sacrococcygeal pain. Preoperative diagnosis can be assisted significantly by CT and MRI scanning that is able to show not only the size of the tumor and whether it is cystic or solid, but also its spatial relationship to adjacent structures (ureter, urinary bladder, rectum, uterus, great vessels), soft-tissue plans, presence or absence of bony invasion and nerve involvement. Diagnostic biopsies are not usually recommended, because they are associated with high rate of complications. Indications for biopsy are unresectable tumors with necessity of diagnosis for the adjuvant therapy and in case of Ewing sarcoma or osteogenic sarcoma where the patient may benefit from neoadjuvant therapy. [51],[52],[53]


 > Treatment Top


Complete surgical resection is the millestone of treatment for sacrococcygeal neoplastic lesions, even if the patient is asymptomatic. Surgery resection establishes the diagnosis, prevents possible malignant degeneration of benign lesions and avoids the possibility of infection of cystic masses. Surgery resection depends on location, size, involvement of sacrum, coccyx and involvement of adjacent visceral structures. When tumors involving sacrum and sacral nerves, a multidisciplinary approach is necessary in order to prevent at least unilateral S3 nerve roots, responsible for normal bowel and bladder function. [54] There are described three approaches for resection of sacrococcygeal tumors: Transabdominal anterior approach, perineal-transsacral posterior approach and abdominoperineal combined approach. [55] With a posterior approach small tumors located below the level of S3 vetrebra can be treated. For tumors located above the level of S3 it is better to consider the transabdominal approach, while for tumors located at the level of S3 with need of sacrococcygeal resection, it is better to use the combined approach. Many authors believe that malignant lesions may be resected with the sacrococcygeal area and when adjacent structures are involved (rectum, vessels) the en bloc resection becomes necessary. Sacral segments may need to be resected either for surgical approach or in order to obtain clear margins. Coccyx must be always resected. Rectal resection rarely must be performed. This en bloc resection, when necessary, may be accompanied by significant perioperative morbidity and functional disability. It is clear that it is in the surgeon's hand to weigh the benefits of such extended resections and it seems that the prognosis of these patients depends on the nature of the lesion and especially on the quality and completeness of the surgical resection. [56],[57],[58],[59]


 > Conclusion Top


Sacrococcygeal neoplastic lesions are considered to be a diagnostic and therapeutic challenge. A lot of awareness and a high index of suspicion is necessary when dealing with chronic sacrococcygeal pain and occult sacrococcygeal masses. Characteristics, location and possible invasions to the adjacent structures or tissues can be evaluated with CT and MRI. Complete surgical resection with possible clear margins is the gold standard of surgical treatment and radiotherapy is suggested almost in all cases of sacrococcygeal tumors post-operatively. Many authors agree that a multidisciplinary approach including colorectal surgeons, neurosurgeons and radiation oncologists could improve peri-operative and post-operative outcome and offer essentially to the quality of life.

 
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