Concomitant Pyogenic Atlantoaxial Spondylodiscitis with Retropharyngeal Abscesses and Tuberculous Spondylodiscitis with Gibbus Deformity: A Combined Rare Condition—A Case Report and Literature Review

Abstract

Concomitant pyogenic atlantoaxial spondylodiscitis alongside retropharyngeal abscesses, in conjunction with tuberculous spondylodiscitis manifesting as gibbus deformity, represents a rare but significant clinical entity. This dual infectious process poses considerable risks and can lead to severe, life-threatening complications if not appropriately managed. We present an atypical case of a 6-year-old Thai boy with concurrent pyogenic atlantoaxial spondylodiscitis, retropharyngeal abscesses, and tuberculous spondylodiscitis at the T6 to T8 levels, leading to a progressive kyphotic deformity. The surgical treatment involved transoral drainage of the abscesses, followed by debridement and vertebral column resection at T6 to T8. A titanium mesh cage was placed, and instrumentation from T3 to T11 was performed using pedicle screws and rods. Postoperatively, the patient showed favorable recovery, with the Cobb angle improving from 70 to 16 degrees. He received intravenous antibiotics for 2 weeks, then oral antibiotics for 4 weeks, along with 12 months of antituberculous chemotherapy. Over a 2-year follow-up period, the patient exhibited clinically significant improvement, and postoperative radiographs confirmed solid osseous fusion with no indications of loss of correction or implant failure. Concomitant pyogenic atlantoaxial spondylodiscitis with retropharyngeal abscess formation, alongside tuberculous spondylodiscitis leading to gibbus deformity, constitutes a rare yet serious clinical scenario. If not addressed promptly, the condition carries substantial risks, such as airway obstruction, sepsis, and potential neurological impairments. Management strategies should prioritize the elimination of infectious agents, prevention of neurological compromise, stabilization of the spinal column, and correction of kyphotic deformities.

 

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Keywords

pyogenic spondylodiscitis - bacterial spondylodiscitis - retropharyngeal abscess - tuberculous spondylodiscitis - gibbus deformity

Introduction

Spinal infections are relatively rare, representing only 2 to 4% of all osteomyelitis infections. The estimated mortality rate ranges from 1 to 20%, depending on the patient group and the infecting agent.[1] In addition, pyogenic atlantoaxial spondylodiscitis with retropharyngeal abscess extension is rare and can be considered a serious complication, such as upper airway obstruction, with mortality rates of up to 2.6%.[2] [3] [4] Meanwhile, tuberculous spondylodiscitis, also known as Pott's disease, was first described in 1779 by Dr. Percival Pott.[5] It accounts for 50% of all cases of skeletal tuberculosis. The most common site is the thoracolumbar junction, followed by the lumbar region. The incidence of neurological complications in tuberculous spondylodiscitis varies from 10 to 43%.[5] [6] As the disease progresses, it destroys spinal elements, resulting in an angular deformity. Delays in diagnosis and treatment may lead to permanent neurological deterioration, paralysis, or even fatalities if left untreated. The combination of these conditions is rare and can cause life-threatening problems if not properly diagnosed and treated. Therefore, the objectives of the study were to report this combined rare condition and to review the literature.

 

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Case Presentation

A 6-year-old Thai boy was transferred and admitted to our institution with a 3-month history of neck stiffness, torticollis, and progressive kyphotic back deformity. He also has difficulty tolerating oral intake and experiences significant discomfort with neck extension, often keeping his neck in a flexed posture. His parents reported an associated medical history of upper respiratory tract infection before this admission, and he had no prior contact with tuberculosis. Upon physical examination, his head was tilted to the left side (torticollis) with a palpable nonmovable soft tissue mass measuring 3 × 5 × 5 cm³ at the right submandibular area extending to the center of the chin and neck, along with enlarged cervical lymph nodes. Facial nerve function remained intact. Additionally, he experienced severe axial backache with progressive thoracic kyphotic deformity and exhibited trace, pain-limited weakness in both hips; however, sensation remained intact, and no myelopathic signs were noted ([Fig. 1]). Plain radiographs of full-length free-standing in posteroanterior and lateral (whole spine) views ([Fig. 2]) and a computed tomography scan of the whole spine ([Fig. 3]) revealed an osteolytic lesion at the atlantoaxial region with the widening of prevertebral soft tissue shadow from the basion to the C4 vertebrae. Moreover, the mid-thoracic region showed severe T6 to T8 vertebral bodies collapse, resulting in kyphotic back deformity (Cobb angle 70 degrees). Magnetic resonance imaging (MRI) of the cervical spine ([Fig. 4]) demonstrated abnormal marrow signal intensity of atlantoaxial vertebral bodies with a large retropharyngeal abscess extending from the basion to the C4 vertebra anteriorly. In addition, an MRI of the thoracic spine with screening of the whole spine ([Fig. 5]) showed significant destruction of vertebral bodies from T6 to T8 vertebrae with prevertebral abscess formation and subligamentous spreading from T5 to T9 vertebrae. Furthermore, this abscess extends from the prevertebral region to the posterior longitudinal ligament, creating an extradural mass effect that compresses the thecal sac. Laboratory findings revealed an elevation of white blood cell count, erythrocyte sedimentation rate, and C-reactive protein. Aerobic culture from a blood sample, Gram, and acid-fast stain from sputum were unremarkable. Blood for QuantiFERON-TB Gold assay test was positive.

Fig. 1 The patient's overall appearance indicated a tilting of the head to the left (torticollis) and a significant kyphotic deformity in the middle of the thoracic region.

Fig. 2 Preoperative plain radiographs of full-length free-standing in posteroanterior and lateral (whole spine) views showed tilting of the skull to the left side with widening of the mediastinal soft tissue shadow (A) and significant kyphotic back deformity at the mid-thoracic spine, measuring Cobb angle was 70 degrees (B).

Fig. 3 Computed tomography (CT) scan revealed an osteolytic lesion at the atlantoaxial region with the prevertebral soft tissue shadow widening from the basion to the C4 vertebrae. The mid-thoracic region showed kyphotic deformity with severe T6 to T8 vertebral bodies collapse (A—C). A coronal view showed a thickening of mediastinal soft tissue at the mid-thoracic area (D).

Fig. 4 Magnetic resonance (MR) imaging of the cervical spine (A and B) demonstrated abnormal marrow signal intensity of atlantoaxial vertebral bodies with a large retropharyngeal abscess extending from the basion to the C4 vertebra (white arrow).

Fig. 5 A magnetic resonance imaging (MRI) of the thoracic spine with screening of the whole spine (A and B) showed significant destruction of vertebral bodies from T6 to T8 vertebrae with prevertebral abscess formation and subligamentous spreading from T5 to T9 vertebrae. Besides, this abscess extends from the prevertebral region to the posterior longitudinal ligament, extending to form an extradural mass effect compressing onto the thecal sac.

The patient underwent two-stage procedures. First, a transoral incision and retropharyngeal abscesses drainage was performed ([Fig. 6]), followed by radical debridement and paravertebral abscesses drainage with vertebral column resection at T6 to T8 vertebrae and filled the defect with titanium mesh cage insertion with fusion and instrumentation with pedicle screws and rod system from T3 to T11 ([Figs. 7] and [8]). The retropharyngeal abscesses were taken and cultured. Aerobic culture revealed numerous Streptococcus pneumoniae. The surgically resected tissue from the thoracic region was sent to the pathology department for a molecular tuberculosis test (GeneXpert), which yielded a positive result. After surgery, the patient recovered well. Postoperative radiographs showed improvement in the correction of kyphotic deformity ([Figs. 8] and [9]). He was continually treated with intravenous antibiotics for 2 weeks and then switched to oral form for another 4 weeks. Antituberculous chemotherapy was administered for 12 months. Over a 2-year follow-up period, the patient demonstrated substantial clinical improvement. Postoperative radiographic assessments confirmed successful solid osseous fusion, with no observed loss of alignment or failure of the implant.

Fig. 6 Demonstrates transoral retropharyngeal abscess drainage (A). Yellowish turbid pus was collected from the procedure (B).

Fig. 7 Shows intraoperative pre- (A) and post-mid-thoracic kyphotic deformity correction (B) with T6 to T8 vertebral column resection and filled the defect with a titanium mesh cage and instrumentation with pedicle screws and rod system from T3 to T11.

Fig. 8 A comparison of pre- (A, C) and postoperative (B, D) plain radiographs of full-length free-standing in posteroanterior and lateral (whole spine) views showed improvement in kyphotic deformity correction (preoperative Cobb angle 70 degrees, postoperative Cobb angle 16 degrees).

Fig. 9 Shows a comparison of pre- and postoperative general appearance of the patient.

 

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Discussion

Retropharyngeal abscesses that expand directly from the atlantoaxial region are rare but potentially life-threatening infections mainly affecting children aged 5 and younger. They are located between the buccopharyngeal fascia anteriorly and the alar fascia posteriorly.[2] [3] [7] Untreated retropharyngeal abscesses can result in upper airway obstruction and potentially lead to asphyxiation. Approximately 50% of retropharyngeal abscesses are associated with prior upper respiratory tract infections, which can lead to retropharyngeal suppurative lymphadenitis and eventual abscess formation.[7] [8] Most common organisms include group A Streptococcus pyogenes, Staphylococcus aureus, Fusobacterium, and Haemophilus, and other respiratory anaerobic organisms.[7] [9] [10] In this particular condition, it is essential to be mindful of the following indicators that may signify an upper airway obstruction: difficulty swallowing (dysphagia), painful swallowing (odynophagia), inability to manage oral secretions, neck stiffness, torticollis, voice changes (such as a muffled voice or nasal-sounding speech), difficulty opening the mouth (trismus), neck swelling, swollen lymph nodes in the neck (cervical lymphadenopathy), chest pain (which could point to mediastinitis), and breathing difficulties (such as noisy breathing, rapid breathing, and visible retractions of the chest wall).[7] When considering the initial intravenous antibiotic therapy regimen, it is recommended to include either ampicillin-sulbactam (50 mg/kg every 6 hours) or clindamycin (15 mg/kg every 8 hours). Intravenous antibiotics should be continued until the patient is afebrile for at least 24 hours. Once patients show improvement and remain without fever, they may transition to the oral form for at least 14 days. Surgical intervention is indicated when antibiotic therapy fails for 24 to 48 hours or when retropharyngeal abscesses are more extensive than 2 to 2.5 cm.[7] [11] [12] The transoral approach is preferred for draining retropharyngeal abscesses due to its ease of incision and drainage, particularly without a skin incision. Following the procedure, patients need to be closely monitored for possible airway complications. The patient underwent continuous treatment with intravenous antibiotics until reaching clinical stability, at which point the treatment was transitioned to an oral form.

Tuberculous spondylitis is the most common type of spinal tuberculosis, accounting for 60%, followed by arachnoiditis (20%), meningitis (12%), and intramedullary lesion (8%).[13] [14] The most common pathogen of tuberculous spondylitis is Mycobacterium tuberculosis, a slow-growing aerobic organism with a growth-doubling time of about 20 hours in conditions favorable to the bacillus.[15] Symptoms are usually slowly progressive and nonspecific, including fatigue, weight loss, fever, and chronic back pain.[16] As the disease progresses, it causes destruction of the intervertebral disc space and the adjacent vertebral bodies. This leads to the collapse of the spinal elements and anterior wedging, resulting in an angular deformity. This deformity, known as gibbus formation, is palpable due to the involvement of multiple vertebrae.[17] This angular deformity causes compression of the spinal cord, leading to serious neurological consequences such as paralysis. The treatment goals are to eliminate the infection, stabilize the spine, and correct the kyphotic deformity. Surgical criteria include cases with an uncertain diagnosis requiring open surgical biopsy, significant motor deficits (less than two out of five muscle strength), rapidly progressing neurological deficits at presentation, and deformity indicating progression or instability (patients with kyphosis greater than 60 degrees and pediatric patients with signs of a “spine-at-risk”).[18]

The cooccurrence of bacterial spondylodiscitis at the atlantoaxial level, accompanied by a retropharyngeal abscess, alongside tuberculous spondylodiscitis manifesting as progressive kyphotic deformity, is an unusual clinical presentation. Previous literature has documented comparable cases; however, these typically involve infections from either the same pathogen or different pathogens at the same anatomical site. Hsu and Chen[19] present a rare case involving a retropharyngeal abscess in conjunction with cervical Pott's disease and a tuberculous abscess of the chest wall. The optimal management approach remains a subject of debate; however, they advocate for the drainage of both the retropharyngeal and chest wall abscesses, followed by a prolonged course of antituberculous therapy lasting a minimum of 9 months. Similarly, Alawad and Khalifa[20] describe a case of a 6-year-old male patient diagnosed with a tuberculous retropharyngeal abscess linked to cervical Pott's disease. This case was managed with percutaneous drainage alongside a regimen of antituberculous medications administered for 6 months. To our knowledge, this is the first report highlighting the simultaneous occurrence of distinct infectious etiologies in such severe cases that necessitate prompt intervention.

In conclusion, this case report highlights the necessity for increased vigilance regarding a rare and severe combined condition characterized by concurrent pyogenic atlantoaxial spondylodiscitis and retropharyngeal abscess formation, along with tuberculous spondylodiscitis resulting in gibbus deformity. Without timely intervention, this complex pathology poses a significant risk for morbidity and mortality. Therefore, the primary treatment objectives should focus on the rapid eradication of the infections, stabilization of the spinal structure, and correction of the associated kyphotic deformity.

 

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Conflict of Interest

None declared.

Ethical Approval

The study was approved by the ethics committee of Lerdsin Hospital.

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Artikel online veröffentlicht:
10. März 2025