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. 2024 Nov 28;126:110681. doi: 10.1016/j.ijscr.2024.110681

Sacral nerve preservation on total sacrectomy of a large atypical sacral chordoma resection: A case report

MNS Budi a, AK Hadar b, D Fachri c,⁎, YR Wendy c
PMCID: PMC11648255  PMID: 39616745

Abstract

Introduction and importance

Chordoma is a slow growing benign cartilaginous neoplasm. It is rarely found in the spine, comprising only 3 % of all Chordomas. Although Chordoma is typically asymptomatic, the mass effect of the lesion on the spinal cord or nerve roots might result in a progressing neurological loss.

Presentation of case

A 39-year-old male patient presented with pain and a mass on the buttock. The patient complained of intermittent pain for three years before hospital admission. The mass was slowly growing from the size of a marble to the size of a ping-pong ball. The patient also complained of difficulty having bowel movements, bladder and erectile dysfunction for the last 5 months. The biopsy test results in a Chordoma at regio gluteus which gave an impression of a Chordoma arising from the sacral region. Huge Sacral chrodoma was planned to be managed with total resection. Right L5 injury occurred during the surgery. The patient was stabilized with lumbopelvic stabilization. The patient could walk with minimal pain and did not show any disturbance in urination and defecation.

Clinical discussion

The mass was then excised with no intraoperative complication. At follow-up, the patient was asymptomatic and neurologically intact. Lumbopelvic stabilization can be indicated as an option for stabilization post total sacral resection because patient can have early mobilization.

The conclusion

In this case shown that even with large mass excision and lumbopelvic stabilization surgery with sacral nerve preservation and show a good clinical and functional outcome.

Keywords: Chordoma, Sacral Chordoma, Total resection;Lumbopelvic stabilization

Highlights

  • •

    This case report details a rare occurrence of a large chondroma located in the gluteal region, which is an uncommon site for this type of tumor.

  • •

    Surgical management involved a total resection of the tumor and lumbopelvic stabilization to address the sacral chordoma and ensure early mobilization of the patient.

  • •

    The report highlights the importance of complete tumor removal and effective stabilization in improving clinical and functional outcomes for patients with sacral chondromas.

  • •

    The case contributes to the limited literature on the management and outcomes of large gluteal chondromas with lumbopelvic stabilization.

1. Introduction and Importance

Chordomas are cartilage-forming tumors that comprise approximately 5 % of all primary bone tumors and are thought to arise from a muscle tendon or synovial sheath [1,2]. Men are twice as likely to have a Chordoma than women, and these lesions typically present during the third to fifth decades of life [3].

Sacrococcygeal chordoma is a rare primary malignant tumor of the bone, and the most primary sacral tumor [4]. Chordomas may remain asymptomatic and are frequently discovered during radiologic examinations, they may manifest as a rigid paravertebral swelling or with pain, less frequently as a fracture, or even less frequently as a slowly developing neurologic syndrome. A late diagnosis may be the primary cause of severe neurologic deficits. The vertebral body, the neural arch (pedicle and lamina), or the processes of the vertebra (transverse processes and spinous processes) may be affected [5]. The tumor are quite invasive, without timely treatment, and may develop metastasis and lead to progression of diseases.

Extraskeletal Chordomas are uncommon benign soft tissue cartilaginous tumors [6]. Their precise aetiopathogenesis remains unknown. It has been proposed that they originate from pleuripotent mesenchymal cells that differentiate along cartilage lines. These tumors most frequently affect the fingers of middle-aged individuals, with the toes and feet also being affected. Rarely, it can affect the parotid gland, the fallopian tube, and the cranium [7]. Aim of this study is to present our experience in managing sacral chordoma. The patient underwent surgical resection. The ethical commission of our university approved this case report. The patient provided written informed consent. This case report was in line with SCARE 2023 criteria [26].

2. The presentation of case

A 39-year-old man from Indonesia came to the clinic with chief complaint of pain while sitting and a gradually increasing mass over the last three years. At first the lump was as big as a marble, then enlarged to the size of a ping pong ball. The patient was previously treated for his condition by a traditional therapist but his clinical condition did not improved. Due to persistent pain and a growing lump, the patient was then taken to Kawaluyaan Hospital and referred to our tertiary hospital.

For the past 5 months, the patient complained of having difficulties with defecation, urination, and erection. There were no other complaints such as nocturnal pain, loss of appetite, dyspnea, lumps in other body regions, or family with similar complaints. The patient experienced any weight loss and showed no sign of tuberculosis.

Physical examination was performed, and vital signs were normal. At the posterior pelvic region, a lump was found on the gluteal fold with a size of 15x15x30 cm. (Fig. 1) There were scars due to the previous biopsy, but there was no shiny skin, venectation, and distal swelling found. The lump was immobile, smooth, ill-defined margin, warm, and no sign of inflammation. On physical examination of lymph node there are no palpable mass. Mobility examinations were performed, and there was a limited range of movement on the hip joint due to the lump on the gluteal fold. On neurological examination, physiological reflexes were positive, and there were no differences in the right and left extremities but on the left patellar and Achilles reflexes were reduced, and pathological reflexes were absent. There was positive urinary and defecation retention, suggesting autonomic disturbance. Laboratory assessment consist of routine haematologic, electrolyte, and fasting blood glucose were normal. On radiologic xray examination revealed soft tissue tumor at sacrococcygeal area with narrowing of discus and intervertebralis vertebra L5-S1. (Fig. 2).

Fig. 1.

Fig. 1

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Presentation of patient. Clinical appearance of the gluteal region: gluteal mass was found.

Fig. 2.

Fig. 2

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Preoperative Sacral plain radiograph with (left) anteriorposterior view (right) Lateral view.

The patient underwent MRI evaluation twice, a year before the MRI evaluation was concluded as suspect sacral Chordoma (Fig. 3). A year later, the MRI evaluation also suggested sacral Chordoma (Fig. 4). the second MRI found a predominantly cystic mixed mass with necrotic components in it, well-defined borders and regular edges, which destruct the sacral os as high as sacral 2 to coccygeal, extending around obliterating parts of the bilateral gluteus maximus musculus, bilateral internal obturator musculus, bilateral piriformis musculus, and coccygeus musculus. The mass begins to attach to the medial musculus portion of the gluteus minimus bilaterally to the anterior appearing to urge the vesical urinaria, prostate, rectum, and intestines to the anterior. Posteriorly, the mass extends from obliterating the soft tissue to the surrounding subcutaneous tissue. During surgery, the tumor's anterior displacement of the rectum and urinary bladder was managed by employing gentle retraction techniques to create a safe surgical field without causing damage. The anterior structures were carefully identified and protected using a combination of preoperative imaging guidance and intraoperative monitoring.

Fig. 3.

Fig. 3

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Findings on the first MRI evaluation: The mass that destructs the os sacral as high as sacral 2 to the coccygeal extending to the inferior obliterates the musculus gluteus minimus medioinferior, urges the muscular obturator internus bilaterally and anus, to the anterior urges the urinary vesicles and prostate to the anterior.

Fig. 4.

Fig. 4

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Cystic mixed mass with necrotic components, well-defined bordered, regular edges, which destruct the sacral os as high as sacral 2 to coccygeal. The mass started to attach to the medial musculus portion of the gluteus minimus bilaterally to the anterior appearing to urge the vesical urinaria, prostate, rectum, and intestines.

To confirm the diagnosis, a histopathological evaluation (core biopsy) was performed. On gluteal core biopsy preparation, there were pieces of tumor mass consisting of round cells and oval shapes that grow hyperplastic, compacted, and were arranged in groups. The nucleus were pleomorphic, hyperchromatic, and mitotic. Cytoplasm appears partly eosinophilic, partly vacuole and formed a physaliphorous arrangement. Stroma connective tissues were partly myxomatous degenerated. The histopathological evaluation was concluded as gluteal Chordoma, and the patient was planned to undergo tumor resection.

After tumor resection and pedicle screw installation, the patient was treated and observed in our inpatient ward.

The Surgery was conducted by the subdivision of spine and oncology of orthopaedics surgery. The goal of surgery is eradication while retaining function as possible. Under general anesthesia, we decided to perform a posterior approach surgery with en bloc resection (remove the entire tumor in one piece together with a layer of healthy tissue). The patient was positioned in a prone position (Fig. 5). The tumor mass was identified from the bottom of sacral 2 to the coccygeal (Fig. 6, Fig. 7). The tumor impinged and attached to right S1 Root. The excision continued by resecting S1 Nerve root from the tumor (Fig. 8) The resection was completed and followed by lumbopelvic reconstruction with fusion of sacroiliac joint (Fig. 9). Spinal stability must be provided using appropriate replacements with a pedicle screw as internal fixation. Xray of lumbosacral before the surgery shows that there was a mass that causing a destruction of os sacrococcygeal and malalignment of vertebra L5-S1 (Fig. 2). On the post operation x-ray shows that there's a posterior stabilization on lumbal dan ilium, there is no mass left that causing a destruction.

Fig. 5.

Fig. 5

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Marking of the anatomy and planned incision area.

Fig. 6.

Fig. 6

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Exposed tumor from (left) posterior view, (middle) lateral view.

Fig. 7.

Fig. 7

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Exposed of the tumor.

Fig. 8.

Fig. 8

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Post tumor excision and laminectomy.

Fig. 9.

Fig. 9

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The installation of the rods pedicle screw (posterior stabilization).

A few days after operation, the patient felt pain on the operation site but the pain was slowly reduces by day. The patient was then discharged. On discharge, the patient was able to move both his legs, with minimal pain sensation but was still unable to walk (Fig. 12).

Fig. 12.

Fig. 12

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Clinical presentation of the patient after the surgery.

The patient was then followed-up in the outpatient setting. The patient was given symptomatic therapy and underwent physiotherapy. The patient was compliant with the treatment plan and made improvement. On the last follow-up, the patient functional condition was improved and proven by the patient did not feel any pain or numbness on the side of operation site and lower extremity. The patient was before only able to stand up without walking even with the help of walking crutches because the patient felt pain and does not have the strength to walk, the patient now able to kneel and walk with a help of a walking crutches. The patient's autonomic function such as defecation and urination were normal.

3. Discussion

Extraskeletal Chordoma, also known as Chordoma of soft tissue, is a benign tumor of soft tissue comprised primarily of hyaline cartilage and unconnected to bone or periosteum [8]. Chordoma is frequently found in men after 60 years old, meanwhile our case was found in a 39-year-old man. It is typically small, measuring less than 3 cm in diameter, but in our study, the size of the lump was particularly large, measuring 15 cm × 15 cm × 30 cm (Fig. 10), and its histology is similar to that of a periosteal or juxtacortical Chordoma, which is located between the bone and periosteum [6]. Extraskeletal Chordoma (EC) accounts for approximately 1.5 % of benign soft tissue tumors [9]. By definition, these neoplasms arise in soft tissue without any connection to the bone cortex, intra-articular synovium, or periosteum (Fig. 11). EC is typically characterized by a slow-growing, enlarging nodule that may produce pain or tenderness, primarily affecting the hands and feet of adult males, with an excellent prognosis and sporadic malignant transformation [10,11]. The radiological presentation of EC is typically non-specific, revealing a well-demarcated, lobulated soft tissue mass with rare central and peripheral calcifications with a ring-like appearance [12]. To prevent recurrences, it is recommended to remove the entire tumor, including the capsule, even though EC has been regarded as a benign tumor [13]. Sporadic cases in the dura, auricle, parotid gland, and fallopian tube have been reported [14]. ECs outside the hands and feet are exceedingly uncommon, thus our gluteal Chordoma case was accounted as a very rare presentation.

Fig. 10.

Fig. 10

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Resected tumor.

Fig. 11.

Fig. 11

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Postoperative plain radiograph.

Preservation of other sacral nerve roots was crucial in maintaining the patient's autonomic functions and quality of life. The decision not to perform a colostomy was carefully evaluated. Despite the tumor's close proximity to the rectum, there was no direct infiltration into the rectal wall, as confirmed by imaging and intraoperative findings. This allowed for the preservation of the rectum and avoided the need for a colostomy. Comprehensive postoperative wound care protocols, including daily dressing changes and prophylactic antibiotics, ensured successful healing without complications such as infection or wound dehiscence. Various surgical techniques and approaches have been developed to balance effective tumor removal with the preservation of neural elements.

One of the primary techniques is en bloc resection, which involves removing the tumor in one piece along with a margin of healthy tissue. This method is particularly effective in reducing local recurrence rates. However, it requires meticulous planning and surgical precision to preserve sacral nerve roots, especially the S2 and S3 nerves, which are crucial for bowel and bladder control and sexual function. Successful preservation of these nerves can result in significantly better postoperative outcomes for patients.

Hemisacrectomy is another approach where the tumor is removed through a sagittal resection of the sacrum. This technique is advantageous in cases where the tumor does not cross the midline, allowing surgeons to remove the tumor with an adequate margin while sparing the contralateral sacral nerves. Studies have shown that this method provides good oncological outcomes and better functional preservation, with patients retaining partial sphincter function and not requiring permanent urinary catheters or colostomies.

Combined surgical approaches, involving both anterior and posterior access, are also utilized based on the tumor's location and extent. These combined approaches can provide better visualization and access to the tumor, facilitating the preservation of sacral nerve roots. The posterior approach initially exposes the sacrum, followed by the anterior approach to resect the tumor and secure adequate resection margins.

Postoperative outcomes are significantly better in patients where unilateral sacral nerve roots are preserved. These patients generally maintain better bowel and bladder function and have a higher quality of life after surgery. For instance, in a study of 15 patients who underwent hemisacrectomy, all retained partial sphincter function, and none required permanent urinary catheterization or colostomies.

Functional considerations are paramount, as the extent of nerve root sacrifice directly correlates with the degree of functional impairment. Higher-level nerve root sacrifices, such as S1, can result in more severe deficits, including loss of plantar flexion and severe bowel and bladder dysfunction. Therefore, preserving these nerves whenever possible is critical for reducing postoperative morbidity.

In conclusion, the preservation of sacral nerves during Chordoma resection is a complex yet crucial aspect of surgical management aimed at maintaining the patient's quality of life. Techniques like en bloc resection and hemisacrectomy, combined with careful surgical planning, can help achieve effective tumor control while preserving neural function. These approaches ensure that patients experience better functional outcomes and lower recurrence rates post-surgery.

ECs are composed of mature chondrocytes associated with a hyaline cartilage matrix that develops in soft tissues, without bone or joint involvement; usually, ECs are located in fingers, hands, toes, and feet [10,11,15]. Less common locations include the forearms, the chest and abdominal wall, the lungs, fallopian tube, and other visceral organs [13,14]. These tumors are described as well-circumscribed ovoid or lobulated shape, gray-white in color. Histologically, EC is composed of hyaline cartilage arranged in lobules associated with fibrosis, ossification, and myxoid areas. A minor subset of ECs displays a preponderance of chondroblasts resembling osseous chondroblastoma with focal or diffuse calcifications [16]. The neoplastic chondrocytes may demonstrate mild nuclear atypia and visible nucleoli; mitotic figures are rarely observed.

Patients with spinal Chordomas typically appear with localized tenderness at the tumor site. As the tumor spreads into the adjacent paraspinous tissues, there may be a palpable mass present [1]. The radiographic manifestations of a Chordoma may be subtle, and a CT scan is typically required to visualize the pathology. A well-circumscribed, lytic lesion without reactive sclerosis may be visible on a plain film [1]. If the tumor is intraforaminal, local deformity may occur, and the neural foramen may be enlarged [18]. A radiolucent, erosive lesion is seen on a CT scan of bone tissue. On a CT scan, cartilage appears as regions of minimal attenuation. There may be stippled calcification patterns [19]. MRI helps distinguish benign from malignant lesions [20]. A cartilaginous tumor that enhances on MRI is more likely to be a chondrosarcoma [2]. Radiologically distinguishing a Chordoma from a low-grade chondrosarcoma can be difficult.

The recurrence rate of the tumor are above 60 % - 64 % for intratumoral resection and 28 % for resection of marginal tumors. Inadequate surgical resection may lead to intraoperative seeding or contamination that leads to recurrence. The definitive treatment of Chordomas was surgical resection [2]. The purpose of resection is to establish a histological diagnosis, prevent sarcomatous degeneration, and preserve neurologic function [2]. Biopsies guided by CT must be performed with caution, as sampling errors can lead to a false-positive diagnosis of a benign tumor [21]. Due to the cytoarchitecture of the tumor, a malignant focus may be confined to a small portion of the tumor. Generally, a total excision of the lesion is advised [22]. Examining the entire specimen histologically for areas of malignant degeneration is essential. The recurrence rate following complete resection of a Chordoma is less than 10 % [2].

From the result of our literature search, we found only one other case of EC in the gluteal region. Parihar et al. reported a case of a 55-year-old female with an EC sized 5x3x3cm in the gluteal region [23]. Their patient was successfully treated with simple excision. In our patient, the EC was significantly larger. Our case was further complicated by the destruction of sacrum and coccyx by the tumor mass, which required additional procedures. In our cases resection involving saccrum can affect the S1 nerve root, motor deficits, and sexual dysfunction. Mun J et al., had operated a 57 year old man with very large sacral chordoma and was successfully remove with singe stage posterior approach and free from reccurence at 5 years follow up without neurological deficit. And the outcome shows there was no motor weakness, but mild hypesthesia on bilateral buttocks developed and patient show no urination problem. [24] Local recurrence rates of sacral chordoma 43 % to 85 % and metastases rates of recurrence 5 %–40 % at 1–10 years. In our cases, The patient was treated with total sacretomy with single stage posterior approach with lumbopelvic fixation. Total sacrectomy was chosen as the case shown malignant and extensive soft tissue involvement with a possible risk lumbopelvic instability after S1-S3 level resection and reccurence.

The benefit of spinal stabilization is early mobilization. This early mobilization allows for the resumption or improvement of motor function as well as quality-of-life parameters. To accomplish maximum stability but also to preserve lumbar motion segments, The anterior column with its mainly weight-bearing properties has to be adequately reconstructed, but attention has to be paid to the tension bend system of the posterior elements. Lumbopelvic fixation seems to be biomechanically superior by transferring vertical load from the illium directly to the lumbar spine. It prevents flexion of the pelvis in bilateral to salvage highly unstable pelvic ring injuries. This stabilization allows the patient to still have the ability to stand and sit properly. [25,26]

In evaluating the outcome of the surgery, we can assess from clinical and radiological examination. S1-S3 resection can damage L4-L5 nerve root that resulting in anal and urinary incontinence. The xray after the operation shows a stable posterior stabilization and there is no mass left that causing a destruction. Sacral Chordomas commonly present as large masses that are difficult to be fully removed and are associated with complications such as surgical wound infections, wound dehiscence, and neurological deficits, including bowel and bladder dysfunction or sciatic nerve and lumbar plexus injuries. Injury recovery time will show significant improvement within the first 18 months. For the incomplete spinal cord injury experience the largest spark during the first 3–6 months. In this patient the mass was successfully removed, the pedicle screw was installed, and no significant complications were observed.

The few days after the surgery the patient felt pain with VAS ranging from 4/10 to 6/10 but the pain was gradually reduced until the patient merely felt minimum pain. The patient was able to stand up early although the patient still was not able to walk due to pain, and felt he has no strength to walk. After a week of follow up, the patient was able to walk with the help of walking crutches, this improvement was due to the stabilization, which allowed early mobilization. The patient also able to kneel and has normal autonomic function such as urination and defecation.

4. Conclusion

In this particular case, we demonstrated that Chordoma can have an atypical location, such as the gluteal region, and can be substantially larger than what is typically reported in the literature. Moreover, although it does not originate from bones, it can destroy the adjacent structures including bones. We hope that our case report can show the possibility of EC destroying bones. We have also shown that even with large mass excision and stabilization surgery through a posterior approach give a good clinical and functional outcome. L5 injury during tumor resection show good outcome since the patient didn't have any problem in lifting his feet and there was no hypoesthesia around leg regio.

Author contribution

M. Naseh Sajadi Budi: Surgeon, Conceptualization, Visualization, Methodology, Writing and Supervision

Abdul Kadir Hadar: Surgeon, Conceptualization, Visualization, Methodology, Writing and Supervision

Dliyauddin Fachri: Asistant, Writing

Wendy Yolanda R: Assistant, writing

Additional information

The patient received an explanation of the procedures and possible risks of the surgery and gave written informed consent. My manuscript does not contain any personal data. Written informed consent was obtained from the patient for publication of this case report and accompanying images A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying image. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical approval

Ethical approval was provided by the ethical committee of Hasan Sadikin General Hospital, Bandung, West Java, Indonesia on Sept 5th 2024 ethical number 1/rsc/OT/IX/24.

Guarantor

M. Naseh Sajadi Budi, MD

Email: mnsbuddi@gmail.com

Address: Pasteur No.38, Pasteur, Kec. Sukajadi, Kota Bandung, Jawa Barat 40161

Funding source

There is no specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Declaration of competing interest

The authors have no conflict of interest to declare.

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