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19 July 2022: Articles  Saudi Arabia

Atlantoaxial Subluxation Secondary to SARS-CoV-2 Infection: A Rare Orthopedic Complication from COVID-19

Challenging differential diagnosis, Unusual or unexpected effect of treatment, Diagnostic / therapeutic accidents, Rare coexistence of disease or pathology

Shaker Barker1ABCD, Rahaf Mujallid2AB, Karim Bayanzay3CDEF*

DOI: 10.12659/AJCR.936128

Am J Case Rep 2022; 23:e936128

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Abstract

BACKGROUND: There are several reports of atlantoaxial subluxation caused by upper respiratory tract infections. Although, there are many known non-pulmonary complications COVID-19 infection, to date there have been no reported cases of orthopedic complications in the peer-reviewed literature. Diagnosis and management of atlantoaxial subluxation is currently limited. Therefore, it is important to explore other methods of identifying and treating patients suffering from atlantoaxial subluxation.

CASE REPORT: Our patient was an 86-year-old man with right-sided neck pain and reduced range of neck motion for the past 6 months, shortly after a mild case of COVID-19. Autoimmune and inflammatory workup was unremarkable. Patient’s symptoms persisted despite 3 weeks of conservative therapy with analgesics, cervical collar, and physical therapy. He received low-frequency kinetically directed impulse wave (al-Kindi wave) treatment administered by the KKT device after 3-dimensional digital X-ray analysis of the atlas. After receiving the treatment over a period of 13 days, patient showed significant improvement in symptoms and follow-up X-rays.

CONCLUSIONS: For patient’s having neck stiffness or pain with COVID-19, it is important to consider atlantoaxial subluxation as a potential cause, especially if the patient requires intubation, as the technique should be adjusted to reduce spinal injury. Atlas X-ray analysis with Spinalytics provides very precise measurements of the atlas in relation to the skull and cervical spine, and improvement in angles were seen before and after treatment. The al-Kindi wave treatment was also effective in reducing the patient’s symptoms and improving cervical X-ray results, but further studies are required for confirmation.

Keywords: Joint Dislocations, COVID-19, Neck Pain

Background

As we continue to grapple with the SAR-CoV-2 pandemic, we are discovering numerous non-pulmonary complications of the virus. Studies have documented that the SARS-CoV2 virus impacts many other organ systems, such as the heart [1], bone marrow [2], kidney [3], liver [4], brain [5], and other organs [6]. However, no orthopedic complications have been reported.

In the adult population, the most common cause of non-traumatic atlantoaxial subluxation is rheumatoid arthritis [7]. However, there are also several reported cases of upper respiratory tract infections causing atlantoaxial subluxation in adults [8,9]. Currently, there are 2 mechanisms which help describe their association. The first theorizes that severe pharyngeal inflammation extends to the parapharyngeal space and then to the transverse ligament of the atlas, and the inflammation weakens the transverse ligament, causing subluxation. In the second mechanism, the infection enters the atlantoaxial joint through a hematogenous route [10]. Although COVID-19 also causes severe inflammation and accesses the bloodstream [11,12], no cases of atlantoaxial subluxation secondary to the COVID-19 virus have been described in the peer-reviewed literature.

In this case, we describe a healthy elderly man who had atlantoaxial subluxation shortly after a mild case of COVID-19. After initial conservative therapy was unsuccessful, the patient improved with wave treatment administered by the KKT device.

Case Report

TREATMENT:

The KKT treatment delivers low-frequency kinetically directed impulses (al-Kindi waves) toward the atlas at frequencies of 16–200 Hz. The purpose of the treatment is to manage chronic pain. As part of the KKT treatment, patients require digital X-rays of the cervical spine from multiple directions (open-mouth, frontal, and lateral) for analysis by the Spinalytics software. The software describes the atlas position in relation to the skull and cervical spine to determine the appropriate vector to apply the al-Kindi waves to offset atlas deviation.

As shown in Figure 3A and 3B, the Spinalytics analysis showed a deviation of the atlanto-occipital angle 24.90° to the right (average 2.37° right), a clockwise rotation of the atlas of 6.95° in the frontal plane (average 1.86° clockwise), and a 5.00° rightward deviation of the atlanto-cervical angle (average 2.20° left). In the coronal plane, the atlas was rotated clockwise by 6.50° (average 2.14° counterclockwise). Average angles were obtained from an unpublished report calculating summary statistics of the Spinalytics database of patients with back and neck pain.

To offset the computed atlas deviation, the stylus of the KKT device was placed 1.5 cm below the tragus of the right ear. As shown in Figure 4A and 4B, the stylus was pointed at a height vector of 0.58 inches at 334°, with an impulse force of 1.5 lbs. The al-Kindi waves were applied in 18 treatment sessions over 13 days. Initially, the patient received 2 sessions daily, until it was gradually reduced to 1 session every 2 days.

OUTCOMES:

After completing the treatment, the patient described significant improvement in pain (4/10) and increased range of motion in all directions, with the most significant improvement in neck extension. The right-sided tilt had also improved significantly.

The patient noted considerable improvement in quality of life, with improvement in sleep, ability to drive, and performing activities of daily living. As shown in Figure 1B, the patient’s neck position improved appreciably.

Repeat X-rays were performed (Figure 2D–2F), which showed improvement in right-sided tilt in the open-mouth view. As shown in Figure 3C and 3D, the Spinalytics analysis showed a reduction of all the angles. The atlanto-occipital angle had shrunk from 24.90° to 11.70° to the right (average 2.37° right), and there was also a reduction in the clockwise rotation of the atlas from 6.95° to 4.15° in the frontal plane (average 1.86° clockwise). The rightward deviation of the atlanto-cervical angle decreased from 5.00° to 4.00° (average 2.20° left). In the coronal plane, the atlas clockwise rotation was reduced from 6.50° to 3.81° (average 2.14° counterclockwise).

Discussion

COVID-19 AND ATLANTOAXIAL SUBLUXATION:

It is difficult to definitively attribute COVID-19 as the cause of atlantoaxial subluxation because there are other known causes that must first be ruled out. The primary differential diagnosis for causes of atlantoaxial subluxation in adults includes traumatic, infectious, inflammatory, and congenital causes [13]. This patient did not have an attributable history of trauma or congenital disease to implicate as the cause of his atlantoaxial subluxation. An inflammatory condition, such as rheumatoid arthritis, is also an improbable etiology, as the patient did not have peripheral joint symptoms or elevated serum markers.

Many bacterial and viral upper respiratory tract infections have been identified as triggers for atlantoaxial subluxation, either through a contiguous inflammatory process or by hematogenous spread [10]. However, there have been no documented cases describing COVID-19 as a possible source of atlantoaxial subluxation. In theory, COVID-19 may be a possible trigger, since its pathophysiology is similar to other known infectious causes of atlantoaxial subluxation. This is the first reported case providing evidence for such a premise.

It is essential to recognize this association, as it may have implications in the inpatient setting. Approximately 80% of critically ill COVID-19 patients require intubation and mechanical ventilation during their care [14]. Due to cervical instability, atlantoaxial subluxation is a predictor of difficult intubation [15]. Therefore, practitioners must adjust the position and technique of intubation, with studies showing a benefit of the protrusion position, to provide more support and extension at the craniocervical junction [16]. Although further inquiry is required, it may be prudent to use such a technique with COVID-19 patients who report neck pain or stiffness.

COMPUTERIZED IMAGING ANALYSIS:

The Spinalytics analysis described in this case study provides very detailed and precise information regarding the rotation and position of the atlas relative to nearby structures. The images are taken on a standardized track with head clamps to ensure consistent head placement to generate reproducible images that can be digitally analyzed. Previous studies have shown this methodology limits inter- and intra-observer variability [17]. As shown in Figure 3A, our patient had greater atlas rotation that average. After receiving treatment, a significant reduction in all the angles was noted.

It is estimated that 40–85% of rheumatoid arthritis patients with neck pain have atlas instability [18], and approximately 10% die of undiagnosed spinal cord or brain stem compression [19]. Since there are preventative procedures available [20], there is a role for the development of more accurate diagnostic tools for the early detection of at-risk patients. Further studies are required to determine if such detailed information can guide treatment or provide prognostic information.

AL-KINDI WAVE THERAPY FOR ATLANTOAXIAL SUBLUXATION:

Unless there is evidence of neurovascular compromise, severe cervical instability, or unremitting pain, atlantoaxial subluxation is treated non-surgically [21]. In our case, the patient had a Type 1 Fielding and Hawkins subluxation without neurovascular symptoms. Most atlantoaxial subluxations secondary to infection improve spontaneously [22]; however, this was not the case for our patient. A subsequent trial of conservative therapy also did not improve his symptoms.

After 6 months of failed conservative therapy, the patient underwent al-Kindi wave treatment administered using the KKT device. The treatment applies impulses directed toward the atlas to offset deviations detected on digitally analyzed X-ray films. Previous studies have shown that the therapy can improve neck pain and restore cervical spine alignment [23,24]. This case shows that after receiving the al-Kindi wave therapy, there was significant improvement in symptoms and functional status, which was corroborated by follow-up imaging and digital analysis of the atlas.

Considering the KKT treatment aims to correct atlas deviation, the KKT treatment may be a rational treatment for reversing atlantoaxial subluxation. In particular, it may be helpful for patients who failed conservative therapy and would like to avoid surgery. Further studies are required to better understand its efficacy and role in the management of atlantoaxial subluxation.

Conclusions

For COVID-19 patients with neck stiffness or pain, it is important to consider atlantoaxial subluxation as a potential cause, especially if intubation is required, as the technique should be adjusted to reduce spinal injury. Atlas X-ray analysis with Spinalytics provides very precise measurements of the atlas in relation to the skull and cervical spine, and improvement in angles were seen before and after treatment. However, further studies are required to determine the clinical significance of this information. The al-Kindi wave treatment was also effective in reducing this patient’s symptoms and cervical X-rays, but further studies are required to determine if it could be a viable treatment for such patients.

Figures

Figure 1.. (A) Image of the patient before treatment, showing the head’s right-sided deviation with torticollis. (B) Images of the same patient after treatment, showing normalization of head tilt. Figure 2.. (A) Lateral X-ray before treatment in extension showing no anterior/posterior subluxation of the C1. (B) Open-mouth X-ray before treatment shows atlas rotation to the right relative to the dens, consistent with a Type 1 anterior/posterior subluxation. (C) Lateral X-ray before treatment in flexion showing no anterior/posterior subluxation of the C1. (D) Lateral X-ray after treatment in extension showing no anterior/posterior subluxation of the C1. (E) Open-mouth X-ray of the patient after treatment shows significant improvement in atlas rotation relative to the dens. (F) Lateral X-ray of the patient after treatment in flexion showing no anterior/posterior subluxation of the C1. Figure 3.. (A) Diagrammatic representation of the patient’s Spinalytics analysis, displaying a rotation of the atlas in posterior coronal view (red) with average angles (gray) before treatment. (B) Diagrammatic representation of the patient’s Spinalytics analysis displaying rotation of the atlas in inferior transverse view (red) with average angles (gray) before treatment. (C) Diagrammatic representation of the patient’s Spinalytics analysis, displaying a rotation of the atlas in posterior coronal view (red) with average angles (gray) after treatment. (D) Diagrammatic representation of the patient’s Spinalytics analysis displaying rotation of the atlas in inferior transverse view (red) with average angles (gray) after treatment. Images are for diagrammatic purposes only, and angles and distances are not accurately illustrated. Figure 4.. (A) 3D model displaying vector of probe (blue) as applied to the patient. Treatment was applied at an angle of 334° and elevated at 0.58 inches. (B) Reference image displaying how the wave treatment is applied to the patient. Images are for diagrammatic purposes only, and angles and distances are not accurately illustrated.

References:

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2.. Bhattacharjee S, Banerjee M, Immune thrombocytopenia secondary to COVID-19: A systematic review: SN Compr Clin Med, 2021; 2(11); 2048-58

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4.. Zhang C, Shi L, Wang FS, Liver injury in COVID-19: Management and challenges: Lancet Gastroenterol Hepatol, 2020; 5(5); 428-30

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7.. Smith PH, Sharp J, Kellgren JH, Natural history of rheumatoid cervical subluxations: Ann Rheum Dis, 1972; 31(3); 222-23

8.. Uğur HC, Cağlar S, Unlu A, Erdem A, Infection-related atlantoaxial subluxation in two adults: Grisel syndrome or not?: Acta Neurochir (Wien), 2003; 145(1); 69-72

9.. Clark WC, Coscia M, Acker JD, Wainscott K, Infection-related spontaneous atlantoaxial dislocation in an adult: case report: J Neurosurg, 1988; 69(3); 455-58

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13.. Yang SY, Boniello AJ, Poorman CE, A review of the diagnosis and treatment of atlantoaxial dislocations: Global Spine J, 2014; 4(3); 197-210

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15.. Mashour GA, Stallmer ML, Kheterpal S, Shanks A, Predictors of difficult intubation in patients with cervical spine limitations: J Neurosurg Anesthesiol, 2018; 20(2); 110-15

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19.. Mikulowski P, Wollheim FA, Rotmil P, Olsen I, Sudden death in rheumatoid arthritis with atlanto-axial dislocation: Acta Med Scand, 1975; 198(1–6); 445-51

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Figures

Figure 1.. (A) Image of the patient before treatment, showing the head’s right-sided deviation with torticollis. (B) Images of the same patient after treatment, showing normalization of head tilt.Figure 2.. (A) Lateral X-ray before treatment in extension showing no anterior/posterior subluxation of the C1. (B) Open-mouth X-ray before treatment shows atlas rotation to the right relative to the dens, consistent with a Type 1 anterior/posterior subluxation. (C) Lateral X-ray before treatment in flexion showing no anterior/posterior subluxation of the C1. (D) Lateral X-ray after treatment in extension showing no anterior/posterior subluxation of the C1. (E) Open-mouth X-ray of the patient after treatment shows significant improvement in atlas rotation relative to the dens. (F) Lateral X-ray of the patient after treatment in flexion showing no anterior/posterior subluxation of the C1.Figure 3.. (A) Diagrammatic representation of the patient’s Spinalytics analysis, displaying a rotation of the atlas in posterior coronal view (red) with average angles (gray) before treatment. (B) Diagrammatic representation of the patient’s Spinalytics analysis displaying rotation of the atlas in inferior transverse view (red) with average angles (gray) before treatment. (C) Diagrammatic representation of the patient’s Spinalytics analysis, displaying a rotation of the atlas in posterior coronal view (red) with average angles (gray) after treatment. (D) Diagrammatic representation of the patient’s Spinalytics analysis displaying rotation of the atlas in inferior transverse view (red) with average angles (gray) after treatment. Images are for diagrammatic purposes only, and angles and distances are not accurately illustrated.Figure 4.. (A) 3D model displaying vector of probe (blue) as applied to the patient. Treatment was applied at an angle of 334° and elevated at 0.58 inches. (B) Reference image displaying how the wave treatment is applied to the patient. Images are for diagrammatic purposes only, and angles and distances are not accurately illustrated.

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923