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. 2016 Jul 7;8(2):162–170. doi: 10.1111/os.12245

Surgical Treatment of Sacral Neurogenic Tumor: A 10‐year Experience with 64 Cases

Wei Sun 1, Xiao‐jun Ma 1, Fan Zhang 2, Wei‐liang Miao 3, Chong‐ren Wang 4, Zheng‐dong Cai 1,✉
PMCID: PMC6584081  PMID: 27384724

Abstract

Objective

To explore the outcomes of surgical treatment of sacral neurogenic tumors

Methods

Between 1 January 2003 and 31 December 2012, data on 64 patients with sacral neurogenic tumors treated with surgery were retrospectively analyzed. The mean age of the 64 cases (35 males and 29 females) was 37.2 years (range, 21–69 years); 38 had neurilemmomas and 26 neurofibromas. Thirty‐four of the tumors involved S 1 and S 2, 11 S 3 or lower, and 19 were single presacral soft tissue masses. Tumors were removed via anterior, posterior or combined anteroposterior approaches. Patients with unstable sacroiliac joints underwent iliolumbar fixation.

Results

Depending on the extent of tumor involvement, one of three surgical approaches was used: a single anterior approach (19 patients), single posterior approach (25 patients), or a combined anteroposterior approach (20 patients). The mean operation time was 3 h (range, 2–6 h) and the mean blood loss 878 mL (range, 400–3120 mL). The mean duration of follow‐up was 58.2 months (range, 24–93 months). These surgeries had the following complications. Three patients had massive intraoperative hemorrhage and posterior back pain and discomfort postoperatively. One patient had intraoperative ureteral injuries requiring intraoperative ureteral catheterization. In two patients, the tumor involved the S 1 nerve roots bilaterally, necessitating their removal, which resulted in obvious lower limb motion and sphincteric dysfunction. In 13 patients with unilateral tumor involvement of the nerve roots of S 1 and lower spinal levels, only the contralateral nerve roots of the S1 and lower levels were preserved; eight of these patients had impaired bladder and bowel function. Posterior incisions failed to heal in 10 patients, secondary wound healing occurred in nine of them and one required a gluteus maximus myocutaneous flap. Three patients developed postoperative cerebrospinal fluid leaks that were and alleviated by waist belt compression bandaging and placing them in the Trendelenburg position. Eight patients developed tumor recurrences postoperatively; pathological examination of the tissue excised in the second surgeries revealed malignant changes in the three patients with neurilemmomas. There were no intraoperative deaths. Rod fractures occurred in three of the 18 patients requiring iliolumbar reconstruction.

Conclusions

The clinical characteristics of sacral neurogenic tumors make them easy to diagnose. The approach to resection should be determined by the location and size of the tumor. Patients with huge tumors may lose considerable blood intraoperatively and a have higher risk rate of postoperative complications.

Keywords: Approach, Neurogenic tumor, Outcome, Sacral, Surgical management

Introduction

Chordomas, giant cell tumors of bone, neurogenic tumors and other tumors can occur in the sacrum, which harbors the distal end of the spinal cord. Sacral neurogenic tumors, which comprise neurilemmomas and neurofibromas, arise from the sacral nerve, grow along the bony neural foramen and extend inside the sacral canal. Benign peripheral neurogenic tumors include peripheral neurilemmomas and neurofibromas, while malignant peripheral neurogenic tumors include malignant neurilemmomas and neurofibrosarcomas1. Some of the tumors are dumb‐bell in shape; because the growth of these tumors is limited by the sacral canal, they are generally small2; however, when they extend outside the sacral canal, they can bloom into huge presacral masses. Because of their unique growth pattern, sacral neurogenic tumors are hard to manage surgically. Alternatives to surgery include interventional perfusion3, radiofrequency ablation4, endovascular embolization techniques, and computed tomography (CT)‐guided iodine‐125 seed implantation. In our previous study, we showed that CT‐guided iodine‐125 seed implantation or gelatin sponge particle embolization are useful means of slowing the development of giant benign sacral neurogenic tumors5. However, complete resection remains the optimal treatment choice in patients with sacral neurogenic tumors6. Given the need to preserve neurological function when possible and the high local recurrence rate, identification of the surgical strategy that achieves optimal outcomes is essential.

Because most neurogenic tumors extend from inside the neural foramen to outside the spinal canal, there are characteristically few symptoms initially7. Additionally, because they are located deep in the pelvic cavity, these tumors are hard to identify, prejudicing early diagnosis. Thus, patients usually have huge masses by the time of diagnosis. These huge presacral tumors are located deep in the pelvic cavity and their relationships with adjacent organs are complex; it is therefore extremely difficult to identify a clear boundary. Generally, tumors located in this anatomical position are of borderline malignancy and unlikely to metastasize; however, the incidence of recurrence is high. During their excision, the rectum, ureter and iliac blood vessels should be adequately freed and secured, making the surgical procedure difficult. In addition, because the presacral venous plexus is susceptible to rupture and hemorrhage, intraoperative hemorrhage can be copious great and difficult to control; also, attempts to preserve neurological function can result in incomplete tumor resection when performed by inexperienced surgeons, leading to residual tumor, a high local recurrence rate, poorer control, and more complications. Prevention of tumor recurrence depends mainly on complete removal of the tumor via an appropriate surgical approaches, possible approaches including single anterior, single posterior and anteroposterior approaches; the approach play an important role in enabling total removal of the tumor.

Because sacral neurogenic tumors have a low incidence, many surgeons are not familiar with the required surgical procedure. In this retrospective analysis of 64 patients with benign sacral neurogenic tumors admitted between January 2003 and December 2012, our aims were as follows: (i) to describe the clinical characteristics of sacral neurogenic tumors; (ii) to determine the appropriate surgical approaches for better removing sacral neurogenic tumor; and (iii) to identify and deal with the common complications of surgical treatment of sacral neurogenic tumors.

Materials and Methods

General Data

Sixty‐four patients (35 males and 29 females) with sacral neurogenic tumors treated in the Department of Orthopaedics of the Shanghai Tenth People's Hospital from 1 January 2003 to 31 December 2012 were enrolled in this retrospective study. Their mean age was 37.2 years (range, 21–69 years). The pathological tumor types comprised 38 neurilemmomas and 26 neurofibromas. The patients’ initial symptoms included lower limb pain in 36 cases, impaired bladder and bowel function in 23 cases, palpable painless masses in the lower abdomen in four cases, perineal numbness in eight cases and positive findings during routine health checks in nine cases.

Preoperative imaging examinations indicated that the tumors involved S1 and S2 in 34 patients, S3 or lower in 11 patients entrenched (Fig. 1) and were single presacral soft tissue masses in 19 cases. In 2009, Wei et al. published a classification of neurogenic tumors arising from the sacrum based on growth pattern and comprising the following four types8. In type I, tumor growth is confined to the sacral canal, which it may enlarge; in type II, tumor growth extends anteriorly out of the sacral neural foramens with formation of a huge presacral lump; in type III, tumor spreads both anteriorly and posteriorly with formation of masses anterior and posterior to the sacrum; and in type IV, tumor growth is confined to the presacral space, no tumor being present in the sacral canal. In the present study, nine patients had type I tumors, 29 type II, seven type III and 19 type IV. The sacrum was involved, there bone destruction around the sacral foramens, which were expanded, and the sacral lamina were attenuated in all 45 patients. Six of the patients with presacral soft tissue masses, had urinary tract obstruction and four rectal obstruction. However, the tumor boundaries were clear in all cases and no organs were invaded.

Figure 1.

Figure 1

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Sacral tumors are classified into two types because of the significant differences between the upper sacrum and lower sacrum in maintaining lumbopelvic continuity. (A) Tumors involves S1 and S2 are defined as type I. (B) Tumors involving S3 or lower levels are defined as type II.

All 64 patients underwent intravenous pyelography, digital subtraction angiography and angio‐embolization preoperatively to ascertain whether the ureters were compressed, determine the relationship between the tumor and iliac vessels, and to embolize the vasculature of the tumor during digital subtraction angiography with the aim of reducing intraoperative blood loss. None of the 64 patients received radiotherapy or chemotherapy, but all were followed up closely.

Surgical Procedures

Different surgical approaches were selected based on the extent of tumor involvement9. Because there is a significant difference between the upper and lower sacrum regarding lumbopelvic continuity, sacral tumors can be classified into two types; namely, type I tumors, which have S1 and S2 involvement, and type II tumors, which involve S3 and lower levels10. However, the extent to which the tumor protrudes presacrally into the pelvic cavity is the key factor that determines the surgical approach. We defined the independent index D (horizontal distance between the tumor's anterior point and the anterior edge of the sacrum on the sagittal plane of an MRI, Fig. 2) and considered this in combination with the growth characteristics of the tumor to decide between a single posterior, single anterior or combined anteroposterior approach. These tumors expand from inside the sacral neural foramens to outside them and form into huge presacral masses; they may grow both anteriorly and posteriorly, creating both pre‐ and post‐sacral masses.

Figure 2.

Figure 2

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Diagrammatic representation of measurement of the D value.

Single Posterior Approach

A single posterior approach is suitable for tumors that are confined to the sacral canal with involvement of the sacral canal or posterior sacrum, or anterior tumors with a D value less than 5 cm complicated by a low sacrum, such tumors being difficult to expose from an anterior approach. A single posterior approach was utilized in 19 patients (Fig. 3).

Figure 3.

Figure 3

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Diagrammatic representation of the single posterior approach.

Single Anterior Approach

A single anterior approach is suitable for tumors with only presacral growth but no involvement of the sacral. A single anterior approach was utilized in 25 patients (Fig. 4).

Figure 4.

Figure 4

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Diagrammatic representation of the single anterior approach.

Combined Anteroposterior Approach

A combined anteroposterior approach is appropriate for type I tumors with presacral masses or type II tumors complicated by anterior masses with a D value greater than 5 cm. A combined anteroposterior approach was utilized in 20 patients (Fig. 5).

Figure 5.

Figure 5

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Diagrammatic representation of the combined anterior–posterior approach.

Marginal resection was performed for benign tumors. In patients undergoing resection via a combined anteroposterior approach, a midline abdominal incision was made via an anterior approach, after which the posterior peritoneum was severed posteriorly to dissect the rectum and expose the tumor. Benign presacral tumors were resected via an anterior approach, gauze being used to pack the dissected and marked margins of the dissected tumor. After closing the abdominal incision, the sacral levels below the levels of tumor involvement were removed to enable complete resection of the tumor and retrieve the gauze. The 18 patients with involvement of more than half the sacroiliac joint required iliolumbar screw‐rod fixation and were able to ambulate after wearing a waist belt for 6 weeks.

Results

Clinical Characteristics

The pathological types comprised 38 neurilemmomas and 26 neurofibromas. The initial symptoms included lower limb pain in 36 cases, impaired bladder and bowel function in 23 cases, palpable, painless, lower abdominal masses in four cases, perineal numbness in eight cases and positive findings during routine health checks in nine cases. Preoperative imaging examinations indicated solitary sacral canal involvement in nine patients, solitary presacral soft tissue masses in 19 patients and both presacral and sacral involvements in 36 patients.

Surgical Data

Depending on the extent of tumor involvement, one of the following three surgical approaches was used: a single anterior approach (19 patients), a single posterior approach (25 patients), or a combined anteroposterior approach (20 patients). For the anterior approach, an approximately 15 cm long incision began below the navel and extended to the superior margin of the pubic symphysis. For the posterior approach, an approximately 20 cm long incision began from the L4 spinous processes and extended to the sacral ridge. Iliolumbar fixation was performed in 18 patients, using a spinal nail and stick fixation system, fixing L4, L5 and the ilium, but with no bone graft. Three patients had implant fractures, two patients 3 years and one 4 years postoperatively and underwent removal of their implants. No patient undergoing reconstruction developed an infection. Drainage tubes were left in situ for 3–7 days postoperatively depending on the volume of drainage: these tubes were removed when the volume was less than 50 mL in 24 hours.

Complications

The mean operation time was 3 h (range, 2–6 h) and the mean blood loss 878 mL (range, 400–3120 mL). The major perioperative complications comprised hemorrhage, adjacent organ damage, nerve damage, infection, and unhealed wounds. The main cause of the massive intraoperative hemorrhage that occurred in three patients was rupture of the internal iliac vein and its branches during the anterior dissection. Some of these patients developed retroperitoneal hematomas that were associated with posterior back pain and discomfort. One patient sustained intraoperative ureteral injuries and was treated by ureteral catheterization, the catheter being removed with the aid of ureteroscopy 3 weeks later. Two patients had tumor involvement of the S1 nerve root bilaterally; these were removed, resulting in pronounced lower limb motion and sphincter of Oddi dysfunction. In 13 patients with unilateral tumor involvement of the nerve roots of S1 and lower spinal levels, only the contralateral nerve roots of the S1 and lower levels were preserved,; eight of these patients had impaired bladder and bowel functions whereas the remaining patients only underwent resection of the involved nerve root. Posterior incisions failed to heal in 10 patients, nine of whom underwent secondary wound healing and one received a gluteus maximus myocutaneous flap. Three patients developed postoperative cerebrospinal fluid leaks that were alleviated by waist belt compression bandaging and placing them in the Trendelenburg position.

Follow up

The duration of postoperative follow‐up was 24–93 months, (mean, 58.2 months). Eight patients developed tumor recurrence; pathological examination of the tissue excised in the second surgeries revealed malignant changes in the three patients with neurilemmomas. There were no intraoperative deaths. Three of the 18 patients requiring iliolumbar reconstruction developed rod fractures.

Discussion

Clinical and Imaging Diagnosis of Sacral Neurogenic Tumors

Sacral neurogenic tumors mainly expand from the neural foramen and grow outside the sacral canal. Neurilemmomas and neurofibromas, the most commonly occurring benign neurogenic tumors, lack clinical symptoms in the early stages11. These tumors usually involve the sacral canal and sacral foramens. The initial symptoms in the present study were lower limb pain (36/64, 56.25%), impaired bladder and bowel function (23/64, 35.9%), palpable painless masses in the lower abdomen (4/64, 6.2%), perineal numbness (8/64, 12.5%), and positive findings during routine health checks (9/64, 14%). The duration of neurological symptoms was often long and apparently unrelated to pain and activities. Some patients had severe tumor pain, particularly during the night. Some patients with huge presacral masses had early bladder and bowel dysfunction. Although the initial X‐ray films showed enlarged sacral neural foramens, this finding is not unique to malignant neurogenic tumors. MRI examinations showed consistent isointensity in most of the benign neurogenic tumors, about 6% with cystic changes; whereas with malignant neurogenic tumors, the MRI intensities were generally heterogeneous and about 75% of the tumors had cystic changes.

Relationship between Sacral Nerve Preservation and Nerve Function

When only the S1 nerve root is preserved, anal sphincter function is lost postoperatively; when the S2 nerve roots are preserved bilaterally, bladder and bowel function can be preserved in 50% of patients; and when an S3 nerve root is preserved unilaterally, most patients retain anal sphincter function12, 13, 14. In this study, two patients underwent resection of the S1 nerve roots bilaterally and had motion dysfunction of the lower limbs and anal sphincter dysfunction postoperatively; prior to surgery one of these patients had nerve pain in the lower limbs and one difficulty with defecation. In 13 patients with unilateral involvement of the nerve roots from S1 and lower levels, only the contralateral nerve roots of the S1 and lower levels were preserved, eight of these patients had bladder and bowel dysfunction. In seven patients, the S2 nerve root was removed unilaterally; two of these patients had bladder and bowel incontinence. Sphincter function was preserved in all the remaining patients.

Maximal preservation of the S1 nerve root is extremely important for motion function, whereas preservation of the nerve roots of S2 and lower levels is extremely important for normal anal sphincter function, thus nerve roots of the S2 and lower levels should be preserved unilaterally whenever possible. In patients in whom it is difficult to preserve nerve roots or those with recurrence that may denote malignant change, adequate margins should be achieved ahead of the palliative preservation of neurological function. Patients with bladder and bowel incontinence can perform exercises to improve sphincter function and some may develop control of bladder function. Ankle fusion or a crutch can be used in patients with motion function impairment. Resection of different levels of sacral nerves leads to different dysfunction.

Selection of a Surgical Approach

Selection of an approach for surgical treatment of sacral tumors is mainly based on the tumor growth patterns8, 15, 16, 17; however, there are currently no well‐established clinical classification systems for planning sacral tumor surgeries either in China or internationally13, 18. We have initiated a discussion about surgical classification of sacral tumors based on advances in surgical treatment of sacral tumors and our clinical experience19. Because there is a significant difference between the upper and lower sacrum regarding lumbopelvic continuity, sacral tumors can be classified into two types; namely, type I tumors, which have S1 and S2 involvement, and type II tumors, which involve S3 and lower levels. The extent to which a tumor protrudes presacrally into the pelvic cavity is a key factor in choosing the surgical approach. We have defined the independent index D (horizontal distance between the tumor's anterior point and the anterior edge of the sacrum on the sagittal plane of an MRI) and combine this with the growth characteristics of a sacral neurogenic tumor to select either a single posterior, single anterior or combined anteroposterior approach. Tumors expand from inside the sacral neural foramen to outside it and form huge presacral masses; they may grow both anteriorly and posteriorly, creating both pre‐ and post‐sacral masses.

A single posterior approach is suitable for tumors that are confined to the sacral canal with involvement of the sacral canal or posterior sacrum or anterior tumors with a D value less than 5 cm complicated by a low sacrum, which makes it difficult to achieve exposure from an anterior approach. A single anterior approach is suitable for tumors with only presacral growth but no involvement of the sacral canal (Fig. 6). A combined anteroposterior approach is appropriate for type I tumors with presacral masses or type II tumors complicated by anterior masses with a D value greater than 5 cm (Fig. 7). The decision on whether to perform an anterior approach depends on the anteroposterior diameter, stage and characteristics of the tumors. For tumors with a large anteroposterior diameter (extent of protrusion into the pelvic cavity, i.e., the D value is greater than 5 cm), type I tumors with presacral masses or malignant sacral neurogenic tumors requiring more extensive resection, an anterior approach is indicated. However, there are also contraindications to an anterior approach. These include patients with histories of repeated and multiple surgeries via an anterior approach or obese patients whose sacrums are located deeply in the body and whose surgical risks and difficulties are therefore high; a single posterior approach is indicated in these patients.

Figure 6.

Figure 6

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A 42‐year‐old man who was diagnosed with sacral tumor based on the initial symptom of perineal numbness and MRI findings. (A) T1 weighted sagittal MRI through the pelvis showing a huge tumor anterior to, but not encroaching on, the sacrum. (B) View of the dissected tumor after resection via an anterior approach. (C) Hematoxylin–eosin (HE) stained section of the resected specimen (×200), (D) S‐100 stain is positive (×100). (E) Vimentin stain is positive (× 100). (F) Cytokeratin stain is negative (×40), all these histological findings indicating malignant neurilemmoma.

Figure 7.

Figure 7

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A 48‐year‐old man who was diagnosed with a sacral tumor based on the initial symptom of lower limb pain and MRI findings. (A) T2 weighted sagittal MRI through the pelvis showing a huge tumor located in S1–5 and encroaching on both the pelvis and posterior sacrum; resection via a combined anterior–posterior approach was performed. (B) Postoperative coronal plane X‐ray film of the reconstruction. (C) Gross view of the tumor after resection via a combined anterior–posterior approach. (D) HE stained section of the resected specimen (×200), (E) S‐100 (×100), (F) epithelial membrane antigen (negative; ×100) and (G) CD34 (positive; ×100), all these histological findings indicating neurofibroma.

One of the 64 patients in this study had S2 involvement, was obese and had a history of surgeries via an anterior approach; therefore, in this case a single posterior approach was adopted with intraoperative attention to protecting the anterior rectum and internal iliac blood vessels bilaterally. Tumor wound and presacral hemorrhage are often hard to control during a single anterior surgery. Ligation of the internal iliac arteries unilaterally or bilaterally can be performed to reduce intraoperative hemorrhage, facilitating determination of the resection margin and decreasing the postoperative recurrence rate. Adequate margins should be achieved whenever possible when excising benign neurogenic tumors, but not at the expense of preserving nerve function. For malignant neurogenic tumors, tumor resection should be adequate and take precedence over palliative preservation of nerve function.

Management of Complications

Complications of sacral neurogenic tumor operation include sever hemorrhage, nerve damage20, 21, damage to adjacent organs, intestinal adhesions, tumor recurrence and failure of posterior incisions to heal22.

Copious perioperative hemorrhage is the primary risk in surgery for sacral tumors15, 23, 24, 25. Techniques for managing intraoperative hemorrhage include internal iliac artery ligation, interventional vascular embolism and lower abdominal aorta block26, 27, 28. Mini‐invasiveness, excellent hemorrhage control, and fewer complications are the principles of management of hemorrhage. If the lesion is located high in the sacrum and involving a large area, abdominal aorta block is the technique of choice because it can reliably control intraoperative hemorrhage. Balloon catheters are minimally invasive and effective for achieving a temporary abdominal aorta block; however, more clinical follow‐up reports are required to confirm their safety and reliability29, 30, 31, 32. In our patients, early application of internal iliac ligation and lower abdominal aorta block performed in the late part of the procedure enabled control of hemorrhage; some patients undergoing single posterior surgeries avoided the trauma of internal iliac artery ligation.

Tumor recurrence remains the commonest postoperative complication during the long‐term follow‐up of sacral neurogenic tumors. Reducing intraoperative hemorrhage, maintaining a clear surgical field and selection of the optimal surgical approach based on adequate preoperative assessment can all facilitate more radical surgery and thus reduce the recurrence rate. In most patients with benign neurogenic tumors, resection can be performed via the enlarged sacral foramens after posterior laminectomy. For patients whose tumors are largely protruding anteriorly and located high in the sacrum, forceful surgery via a posterior approach may lead to residual tumor or rupture/hemorrhage of the internal iliac vein, increasing the chances of local recurrence. Excellent surgical margins are the key to reducing the incidence of recurrence.

Posterior incisions failed to heal in 15 patients, nine of whom achieved secondary wound healing after debridement, whereas one received a gluteus maximus myocutaneous flap. The high incidence of wound complications is associated with impaired nutrition and blood supply to the flap and nerve caused by the major and extensive surgical trauma. Additionally, hematomas may form from hematoceles in the massive cavity remaining after local tumor resection and as a result of poor postoperative drainage. Also, because the incisions are near the anus, they may become infected as a result of contamination by feces. Severe deep infections can be caused by intestinal leaks arising from dissection of necrotic and ischemic rectums or from direct injuries during tumor resection. Intraoperative protection of the rectum and postoperative occlusions and dressing changes of the wounds are therefore critical to the management of infectious wound complications. Our experience suggest that maintaining a patent drainage tube while applying a negative aspiration device to well occluded wounds and then changing the dressings after 5–7 days helps in avoiding complications.

Reconstruction of Iliolumbar Stability

Iliolumbar reconstruction is required in patients with tumor involvement of more than 50% of their sacroiliac joints33, 34. In this study, iliolumbar reconstruction was performed in patients with tumor involvement of the upper sacral segments (S1 and S2). Whether to perform reconstruction to achieve iliolumbar stability remains controversial. Based on our experience, young patients require early postoperative functional exercise, we therefore recommend reconstruction to achieve iliolumbar stability in patients with a long anticipated survival (Fig. 7). We do not recommend reconstruction in older patients or those in whom the condition of soft tissue is poor, because such patients are at high risk of postoperative infection; in these patients, crutches can be used after 8 weeks in bed. Although the surgical scars can limit spinal sinking, patients can then walk upright and there is little impact on their spinal stability. In the present study, 18 patients underwent reconstruction to achieve iliolumbar stability by the technique of Blatter et al. 35, which involves no bone grafting. Three of these patients subsequently had implant fractures, two 3 years and one 4 years postoperatively; all fractured implants required removal. None of these patient developed infection.

Conclusions

The clinical characteristics of sacral neurogenic tumors are well‐known. Selection of the resection approach should be based on the location and size of the tumor. Patients with huge tumors have a high risk of complications.

Grant Sources: This article was supported by Youth Scientific Research Foundation of Health Bureau, Shanghai, China (NO. 20124Y133).

Disclosure: The authors declare they have no conflicts of interest related to this article.

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