CASE REPORT |
https://doi.org/10.5005/jp-journals-10001-1561 |
The Surgical Management of Oral Cancer in a Patient with Advanced Ankylosing Spondylitis: A Case Report
1Department of Surgical Oncology, Cancer Centers of America (CCA), Nashik, Maharashtra, India
2Department of Radiation Oncology, Cancer Centers of America (CCA), Nashik, Maharashtra, India
3Department of Anesthesiology, Cancer Centers of America (CCA), Nashik, Maharashtra, India
4Department of Radiology, Matrix Diagnostic Centre, Nashik, Maharashtra, India
5Department of Plastic Surgery, Cancer Centers of America (CCA), Nashik, Maharashtra, India
Corresponding Author: Harsha Kolgunda Nagappa Setty, Department of Surgical Oncology, Cancer Centers of America (CCA), Nashik, Maharashtra, India, Phone: +91 9113695259, e-mail: drharshakn@gmail.com
Received on: 13 February 2024; Accepted on: 10 March 2024; Published on: 06 September 2024
ABSTRACT
Surgical management of oral cancer in a patient with severe comorbidities can pose many difficult challenges to the surgical team. A 48-year-old male patient who was suffering from long-standing, advanced ankylosing spondylitis (AS) presented with oral cancer. On evaluation, the tumor in the left buccal mucosa was found to be keratinizing squamous cell carcinoma of stage IVA [American Joint Committee on Cancer (AJCC)–tumor, node, metastasis (TNM) staging] and was planned for surgical resection. Due to AS, the patient’s trunk and neck movements were severely restricted, along with trismus and restricted jaw movements. Surgical intervention under general anesthesia in such patients is associated with a substantial risk of spinal injury. The trismus and fixity of the neck rendered an exceedingly difficult surgical position for both the anesthesiologist and the head and neck surgeon. The anesthesiologist was successful in establishing a secured airway by performing awake bronchoscope-guided nasotracheal intubation. The neck of the patient had to be supported by pillows instead of a routine neck extension position used for neck dissections. Wide excision of the entire tumor with bite resection and left-sided modified neck dissection was conducted without any positional change of the neck. Reconstruction was conducted with the left anterolateral thigh flap. There were no surgery-related complications, and the healing of the surgical wounds was satisfactory. The patient was discharged from ward care after 8 days. The pathological staging was pT3N3b, and starting from the 5th week of postsurgery, the patient received adjuvant chemoradiation over 6 weeks. Despite many difficulties that were encountered during management, the patient was able to withstand and recover well. Ankylosing spondylitis (AS), as a comorbidity in head and neck cancer cases, can pose significant challenges to the surgical team. Although previous surgical experience and skills matter, good preoperative preparation and adapting the techniques to the needs of the patient will help in achieving surgical goals, prevent any complications, and ensure a good outcome for the patient.
How to cite this article: Kolgunda Nagappa Setty H, Patil R, Patil N, et al. The Surgical Management of Oral Cancer in a Patient with Advanced Ankylosing Spondylitis: A Case Report. Int J Head Neck Surg 2024;15(1):21-28.
Source of support: Nil
Conflict of interest: None
Patient consent statement: The author(s) have obtained written informed consent from the patient for publication of the case report details and related images.
Keywords: Ankylosing spondylitis, Buccal mucosa, Case report, Cervical spine, Oral cancer, Surgical resection.
INTRODUCTION
Surgical resection of oral cancer can be associated with many challenges, and every case can pose a new challenge to the operating head and neck surgeon. Occasionally, a patient with oral cancer can have a rare comorbidity, and the treating surgeon has to face a never-experienced challenge, adapting his techniques to the demands of the case. Here, we are presenting a case in which a patient with oral cancer was also suffering from long-standing, advanced ankylosing spondylitis (AS). We are discussing the challenges that were faced in the surgical management of the case, along with a review of the literature.
Ankylosing spondylitis is a chronic, progressive, inflammatory, debilitating disease affecting primarily the spine and sacroiliac joints, with a prevalence of 0.9% among the general population. Its etiology and pathogenesis are not yet fully understood, and there is no definitive curative treatment at present.1 Advanced cases of AS patients will have severe restriction of the trunk and neck movements due to the rigid fixity of the entire spine, which is classically described as a “bamboo spine.” These patients are at elevated risk of spinal injury, and injury to the cervical spine can result in quadriplegia.2 Hence, performing any surgery under general anesthesia in such patients is challenging.
CASE DESCRIPTION
A 48-year-old male patient came to our hospital in August 2023 with complaints of an ulcerated growth on the left side of the buccal mucosa. This painless lesion was noticed by the patient 30 days back, that kept gradually increasing in size. The patient was also suffering from a stiff spine with restriction of trunk and neck movements for >20 years and was diagnosed to be having AS. For the past 5 years, he also had restricted mouth opening and difficulty in chewing. Albeit restricted, slow movements, the patient was able to perform all his daily activities. He was not on any routine medication and was occasionally using nonsteroidal anti-inflammatory drugs.
On examination, the patient had a very stiff spine and limited mobility of the trunk and neck. The patient had exaggerated thoracic kyphosis. The neck was straightened and fixed in a slightly flexed position. Mouth opening was restricted with a maximum interincisal distance of 15 mm. There was an ulcer-indurated growth in the left buccal mucosa of size 3.5 × 3 cm, involving almost the entire visible left buccal mucosa with induration extending to the left oral commissure. The lesion was palpable underneath the left cheek skin, suggesting tumor extension to subcutaneous tissues. There was no gross tumor extension to retromolar trigone or alveolus. The buccal mucosa on the right side was normal. In the neck, there was a firm-to-hard nontender palpable enlarged lymph node mass of size 2 × 2 cm suggestive of neck metastasis (Fig. 1). Though the patient did not have a history of hypertension, he was found to have raised blood pressure on several instances during examinations. He had varicosity in both lower limbs. The patient was obese, with a body mass index of 35, and had a short neck.
Figs 1A and B: (A) Preoperative photograph of the patient; (B) Slough-covered ulcer-indurated lesion in left buccal mucosa seen through the limited mouth opening
Before visiting us, the patient had already consulted another doctor who, on suspicion of malignancy, had taken a biopsy of the oral lesion along with ordering a computed tomography (CT) scan of the oral cavity. With the biopsy report stating the lesion to be keratinizing squamous-cell carcinoma, the patient was referred to us for cancer-specific management. The CT scan report stated an ill-defined infiltrative mildly enhancing tissue mass lesion on the left side of the buccal mucosa, measuring 26 × 31 mm in craniocaudal and 20 mm in maximum depth. It involved both superior and inferior gingiva-buccal sulci and infiltrated subcutaneous fat. It extended close to the margins of the upper and lower lips without involving them, RMT was spared, and there was no obvious bone erosion. Multiple, enlarged, heterogeneously enhancing lymph nodes were noted at left level 1-B, the largest measuring 26 × 17 mm (Fig. 2). There was loss of cervical lordosis with calcification of anterior-longitudinal ligament in the cervical and dorsal spine. The diffuse syndesmophytic ankylosis of the cervical spine, along with the ankylosis of sternocostoclavicular and costovertebral bony ankylosis, suggested the advanced state of AS (Figs 3 and 4). The magnetic resonance imaging of the patient also showed the classical features of an advanced state of AS, such as a “bamboo spine” and ankylosed bilateral sacroiliac joints. In the lower dorsal spine, an Andersson lesion (inflammatory spondylodiscitis) was also noted (Figs 5 to 7).
Figs 2A to C: (A) The CT axial; (B) Coronal images show heterogeneous enhancing soft tissue thickening along the left buccal mucosa (thin white arrows), with mild irregularity along the mucosal surface due to ulceration, suggestive of carcinoma buccal mucosa; (C) Axial CT image shows enlarged, heterogeneously enhancing lymph nodes (thick black arrows) in the left submandibular region, suggestive of nodal metastases
Fig. 3: The CT sagittal (S) and coronal (C) images of the cervical spine show diffuse syndesmophytes ankylosis of the cervical spine (bamboo spine), suggesting an advanced state of AS
Fig. 4: The CT axial and coronal images showing sternocostoclavicular (white arrows) and costovertebral (black arrows) bony ankylosis due to the advanced stage of AS and resultant narrowing in the region of thoracic inlet
Fig. 5: T2 weighted (T2W) sagittal and postcontrast T1-weighted with fat suppression (T1-FS) sagittal image of the whole spine showing vertebral body squaring (thin white arrows) with diffuse syndesmophytic ankylosis—bamboo spine (curved white arrows). The fatty signal is noted in the lumbar intervertebral disks, secondary to discal ossification (thick white arrows). Inflammatory spondylodiscitis (Andersson’s lesion) is also seen in the lower dorsal spine (white arrowhead)
Figs 6A to E: The MRI images of the spine. (A) T2Wt sagittal image—the visualized vertebrae show hypointense marrow due to osteopenia with anterolateral bony fusions due to ossification of all and fused syndesmophytes (black arrows); (B) Short tau inversion recovery (STIR) sagittal image; (C) Postcontrast T1-FS sagittal image; (D) T2Wt axial image; (E) Postcontrast T1-FS axial image. Reduced vertebral heights and marrow inflammation/edema in D10 and D11 vertebral bodies (thick white arrows) with deformed vertebral morphology (image D), causing a focal gibbus. Extensive inflammatory soft tissue around these vertebrae (white arrowheads)—compressing bilateral exiting nerve roots and dural enhancement (short white arrows) from D9 to D12 vertebra. In the present case of AS, with no clinical signs/laboratory markers of infection, these findings show chronic inflammatory spondylodiscitis (actually known as Andersson’s lesion)
Fig. 7: The STIR coronal image showing near-complete ankylosis of bilateral sacroiliac joints, secondary to long-standing bilateral sacroiliitis
The patient was diagnosed with oral cancer, that is, left buccal mucosa carcinoma T2N2bM0 Stage IVA [American Joint Committee on Cancer (AJCC)–tumor, node, metastasis (TNM) staging 2017], and surgical management was planned after discussing in the tumor board. The patient consented to the surgery with full awareness of all the risks involved. The preanesthesia evaluation involved neurosurgical and cardio-physician fitness for the surgery. The patient was posted for surgery under general anesthesia. Precautions were taken to avoid any undue spinal injury while shifting and positioning the patient for surgery. Bronchoscopy-guided awake nasotracheal intubation could be performed, and thereby, a difficult tracheostomy was avoided. In the supine position, the neck of the patient had to be supported with comfortable pillows to avoid hyperextension (Fig. 8).
Figs 8A and B: (A) Intraoperative patient’s positioning—neck is supported in the existing natural position with pillow and head ring; (B) The tumor extent at cheek skin, as felt on palpation (red arrow) and skin incisions markings (blue thin arrow)
After securing the airway, the tumor was again assessed. A circumferential incision in the left cheek skin, including oral commissure to get the margins around the tumor, was taken, and a vertical limb from this incision continued inferiorly to meet a horizontal upper neck crease line incision. Although there was fixity of temporomandibular joint (TMJ) movement with significant trismus, intraoral mucosal incisions for conducting marginal mandibulectomy with upper alveolectomy [‘bite resection’] were possible, and the tumor was removed in toto along with cheek skin island. A modified Neck dissection was performed with the removal of all the fibro-fatty lymphatic tissues from level I to V, preserving the internal jugular vein, sternomastoid muscle, spinal accessory, and greater auricular nerves (Fig. 9). The reconstruction of the defect was performed by a free anterolateral thigh flap anastomosing with facial vessels.
Figs 9A and B: (A) Intraoperative photograph after wide excision of the tumor and neck dissection is completed. DG, digastric muscle; IJV, internal jugular vein; M, mandible; Mx, maxilla; SA, spinal accessory nerve; (B) Excised tumor specimen
Although the patient had labile hypertensive calls during surgery and in the immediate recovery period, there were no complications. The patient recovered well in the postoperative period; he was shifted out of the intensive care ward on the second postoperative day and was in general ward care for 1 week. At the time of discharge on the 8 postoperative day, the drains had been removed, the patient was ambulatory, and oral feeding had been started in consultation with a dietician. The patient was followed up with regular dressings, and the sutures were removed after 2 weeks. At the end of 4 weeks, there was good progression in the healing, the operated areas were well closed, and the patient was highly satisfied (Fig. 10).
Figs 10A and B: Postoperative photographs of the patient. (A) Taken after 7 days of surgery; (B) Taken at 1 month after surgery
In the histopathology report, the specimen was found to have an ulceroproliferative tumor of the left buccal mucosa, which was a moderately differentiated squamous carcinoma. The tumor was measuring 3 × 2.5 × 2 cm with a maximum depth of invasion of 2 cm. It was invading subcutaneous tissue, but the underlying bone was not involved. Lymphovascular emboli and perineural invasion were seen. All the cut margins were at least 1 cm away from the tumor. There were metastatic deposits in six out of 28 lymph nodes dissected, and all five lymph nodes in level I-B had an extranodal extension. The pathological staging of the disease stood at pT3N3b. After discussing again in the tumor board patient was offered adjuvant chemoradiation, which the patient started receiving from the 5th week of postsurgery. The radiation oncologist and his technologist encountered difficulties in positioning the patient and making an orfit for him. They made a customized neck rest for the patient. The patient received adjuvant radiation to the tumor bed and the neck nodal region to a dose of 60 Gy in 30 fractions over 6 weeks (five fractions per week) along with concurrent once-weekly cisplatin, and there were no unplanned gaps. The patient tolerated the therapies and recovered well. On follow-up after 5 months posttreatment, the patient was free of disease, back to his routine activities and his job as a shopkeeper (Fig. 11).
Figs 11A and B: (A) Postoperative photographs of the patient taken after 4 months of follow-up; (B) Good intraoral healing
DISCUSSION
The comorbidities associated with patients with oral cancer will increase the difficulties associated with the management of oral cancer. Whereas systemic comorbidities, such as uncontrolled diabetes, hypertension, and chronic renal or liver disease, pose additional challenges during the entire management process, certain localized anatomical problems such as trismus, obese short neck, or postradiation therapy fibrosed areas will add to the challenges faced by both surgical and anesthesiology team. The surgical teams managing cancer patients would have usually gained experience in managing the commonly seen comorbidities, but sometimes there can be a presentation of never-experienced rare comorbidity, and the surgical team must adapt to the new challenges.
Ankylosing spondylitis (AS) is a chronic, slowly progressive, inflammatory, debilitating disease affecting primarily the spine and sacroiliac joints. Its overall worldwide prevalence is 0.9%, with wide variation among different populations. It is one of the common rheumatic diseases and a prototype of the spondyloarthropathies, which share common clinical and genetic features.1,3
The etiopathogenesis of the disease is not completely understood; no single agent or event has been identified as the cause of the disease. Both genetic and environmental factors may have a role in the etiology, with immune-mediated mechanisms implicated in pathogenesis.1 Familial aggregation of the disease and its strong association with human leukocyte antigen B27 (HLA-B27) suggests the genetic basis.4 The corelationship with reactive arthritis and inflammatory bowel diseases suggests that enteric bacteria may play a part in triggering the immunological reactions.5,6
The inflammatory histology, raised serum levels of immunoglobulin A (IgA), low tumor necrosis factor (TNF), low interferon γ, and high interleukin 10 levels are the features of AS.7 The characteristic initial histopathological findings in AS are enthesitis, synovitis, osteitis, and myxoid subchondral bone marrow changes. The chronic granulomatous inflammation in the subchondral tissues with the resulting destruction of adjacent articular tissues is followed by varying degrees of fibrous scarring, woven bone, and new cartilage formation.8 These cartilages eventually ossify, leading to chondral fusion and ankylosis of joints. In the spine, the junction of the annulus fibrosus of the disk cartilage and the margin of the vertebral bone become fused, resulting in a single column of fused spine.9
The AS is more common and more severe in men than in women. The disease is insidious in onset and commonly starts in the second or third decade of life.10 It has a prolonged course, and the definitive diagnosis of AS is often delayed.11 The sacroiliac joints and spinal joints are the most commonly involved, but other joints, predominantly of lower limbs, are also involved. There are several extraarticular manifestations of AS, including uveitis. The initial symptoms of patients are insidious onset dull pain in the back, deep in the buttock or lower lumbar regions which is accompanied by morning stiffness. These symptoms gradually become bilateral, more persistent, and are accompanied by great restriction of mobility of the involved joints.12 Neck stiffness and temporomandibular ankylosis are seen in advanced disease. In severe cases, patients’ posture gets affected by the loss of lumbar lordosis, exaggerated thoracic kyphosis, atrophic buttocks, and forward stooped neck.1
The radiological features of early sacroiliitis are blurring of the cortical margins of the subchondral bone, erosions, and sclerosis, which are better detected by MRI.13 As the disease progresses, the changes become symmetric; the joint space appears wider initially but later gets obliterated by fibrous and bony ankylosis. CT or MRI are useful tools to diagnose the disease and for the monitoring of progression, but the radiographic findings do not always correlate well with disease activity. There is no laboratory test to diagnose AS, but tests for HLA-B27 may be useful in patients with inflammatory back pain, as HLA-B27 positivity is associated with a 16 times greater risk for AS than the general population.1
The management of AS, at present, is only palliative; NSAIDs, along with physiotherapy, form the mainstay of treatment in symptomatic patients, and anti-TNF therapy can be used in patients with active disease. The second-line treatments employed are local corticosteroids and disease-modifying antirheumatic drugs (DMARDs), with limited benefit.14 The disease has a severe debilitating effect on patients, and psychosocial support also forms an important part of the management.
The most important complication associated with AS is spinal fracture, which can happen even with a trivial trauma and sometimes even without any recognizable trauma. The lower cervical spine is the most common site for injury and hyperextension is the most observed mechanism that causes it.2 Andersson lesion, a well-known complication in patients with AS, is the development of localized vertebral or discovertebral lesions of the spine. It may result from inflammation or stress fractures of the complete ankylosed spine and the mechanical stresses prevent the lesion from fusion to provoke the development of pseudarthrosis. The lesion can be completely asymptomatic or can give rise to unbearable pain, a progressive kyphotic deformity, or neurological deficits.15 Aortic insufficiency, with possible congestive heart failure and cardiac conduction disturbances, can also occur in patients with AS.16
Like many other autoimmune diseases, patients with AS can also be at a higher risk of malignancies.17 A meta-analysis by Deng et al. stated that AS is associated with a 14% increase in the overall risk for malignancy with a specific increased risk for digestive and hematological malignancies. They also found AS patients from Asia are at the highest risk for malignancy overall.18 Sun et al. conducted a population-based retrospective cohort study in Taiwan. They found that AS patients have a significantly increased risk of overall cancer, especially for the 50–64-year age-group. Incidence of lung cancer and head and neck cancer risks were significantly higher than in other sites in their study.19 A similar study was conducted by Chan et al. in Taiwan and demonstrated that patients with AS are at an increased risk of cancer, particularly melanoma, prostate, thyroid, bone cancers, and hematological malignancies, but found decreased risks for esophageal and oral cancers in patients with AS.20 However, Kelty et al., in a similar study in western Australia, found no significant increase in cancer risk in AS following adjustment for smoking status and common comorbidities but found that AS patients with cancer had a 37% increased 5-year cancer mortality than those cancer patients without AS.21
When patients with AS need surgical management, both the operating surgeon and anesthesiologists have to face many critical challenges. The surgical team should take high precautions while shifting and positioning the patient, especially when the patient is under anesthesia, as any inadvertent movement that affects the fixed spine can result in spinal fracture and cord injury. Endotracheal intubation in a patient with the fixity of cervical spine and TMJ is a challenging task, and the anesthesiologist must rely on his experience and skills to perform awake bronchoscopy-guided nasoendotracheal intubation.22 If the anesthesiologist does not succeed, the alternative to establish an airway is to perform a tracheostomy in an awake patient. In a fixed forward stooped neck of an AS patient, without the necessary extension, most of the cervical trachea is drawn inside the thorax and lies deeper. The surgeon has to pass through the thyroid tissue and negotiate great vessels to reach the trachea and it becomes more difficult in a short and obese neck. This is dangerous, with a substantial risk of hemorrhage, and should be performed only by an experienced head and neck surgeon.
In oral cancer surgery, TMJ ankylosis causing trismus will impede access, and assessment of tumor and routine trismus-releasing procedures employed in submucous fibrosis will not be helpful. When osteotomies are needed, the use of a powered bone saw with meticulous fine cuts is preferred over using a mallet and osteotome to avoid undue stress on the spine. Neck dissection in a fixed obese neck is again a challenging task and will test the skills and experience of the head and neck surgeon.
In a patient with AS, the anesthesiologist has to actively look for any restriction of chest expansion, cardiac conduction disturbances, or anemia before surgery. In our case, the patient did not have any of the mentioned but had varicose veins in both lower limbs; hence, an osteocutaneous free fibula flap was not an option for the reconstructive surgeon. The laboratory parameters like increased levels of C-reactive protein and erythrocyte sedimentation rate are seen with AS and may give a confused picture in the postoperative period if being evaluated for conditions such as sepsis.23
Raghuram et al. in 2007 reported a case of a patient with AS who underwent a laparoscopic nephrectomy for a left kidney tumor. They discussed the surgical and anesthetic challenges faced in laparoscopic abdominal surgery on a patient with AS.24 We have not come across any study in literature where the challenges that are faced in the surgical management of head and neck cancer in a patient with AS are described.
We also would like to state that one of the crucial factors in our successful surgical management of the patient was that he had good family support, which was evident throughout the course.
CONCLUSION
Ankylosing spondylitis (AS) as a comorbidity in head and neck cancer cases can pose significant challenges to the surgical team. Although previous surgical experience and skills matter, good preoperative preparation and adopting the techniques to the needs of the patient will help in achieving surgical goals, prevent any complications, and ensure a good outcome for the patient.
ORCID
Harsha Kolgunda Nagappa Setty https://orcid.org/0000-0002-7713-351X
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