Cervical spinal manipulation is a common practice, which is performed by either professional or non-professional personnel, owing to its relatively few invasive treatments. It is defined by the International Federation of Orthopedic Manipulative Physical Therapists (IFOMPT) as: “A passive, high velocity, low amplitude thrust applied to a joint complex within its anatomical limit with the intent to restore optimal motion, function, and/ or to reduce pain” [
13]. The known complications resulting from cervical spinal manipulation include cerebral stroke from local pressure on the blood vessels, ligament injury or fractures from excessive pressure or rotation, and spinal cord injury [
4,
14]. After cervical spine manipulation, the estimated incidence of serious adverse events ranges from 1 per 50,000 to 1 per 5.85 million manipulations [
2]. According to reports that the likelihood of injury following spinal manipulation was increased among patients with a chronic coagulation defect, inflammatory spondylopathy, osteoporosis, aortic aneurysm and dissection, or long-term use of anticoagulant therapy [
15]. Epidural hematoma is a disease in which blood accumulates in the epidural space of the vertebral body. This disease is usually caused by trauma or iatrogenic surgery, and may be associated with blood coagulopathies, neoplasms, or degenerative spinal disease. Reports of epidural hematoma caused by neck manipulation are rare, with less than 10 cases reported [
3‐
10]. Most cases occurred in the cervical spine, not in the thoracic or lumbar spine. Some of these cases had cervical spondylosis or a history of oral anticoagulants. Table
2 summarizes the cases of epidural hematoma after neck manipulation [
3‐
12]. In most cases, the location of the hematoma was either posterior or posterolateral. The hematoma in our case was located at the right posterior side. The pathological mechanism of spinal epidural hematoma remains unclear. The mechanism of spinal epidural hematoma might be the same as that of intracranial epidural hematoma [
16]. However, some researchers believe that spinal epidural hematoma is caused by injury of the epidural venous plexus or a sudden increase of venous pressure [
16]. The incidence of spinal epidural hematoma is higher in patients with coagulation disorders and in those taking anticoagulants [
17]. In a case reported by Whedon et al. [
5], the patient had to take coumarin for a long time because of atrial fibrillation and showed stiffness after neck manipulation. Subsequent laboratory tests showed abnormal coagulation function. The results of coagulation-related examinations in this case were normal and there was no history of taking anticoagulants (Table
1). Heiner [
9] reported another interesting case in which the patient did not have the above-mentioned risk factors. However, the patient was pregnant at that time. Because of the change in venodynamics and a decrease in venous pressure in the epidural space relative to venous pressure, the pressure gradient of epidural vessels increased, which easily led to epidural hematoma [
18]. We observed that most of these cases report scarcely description of the clinical characteristics which are possible risk factors for serious complications in patients, such as smoking, cervical trauma, recent infection, hypertension, etc. (This is where our cases are limited) It could be that the manipulating professionals did not see the need to report or were unaware of these items or were more focused on the treatment strategy and recovery after hospitalization [
13]. The CARE statement was published to guide transparency and accuracy of case reports as well as to improve the quality of case reports [
19,
20].
Table 2
Summary of reported cases of cervical epidural hematoma after spinal manipulation therapy
| 33 | Female | Paraplegia | C4–6 | posterior | Surgery | |
| 67 | Female | Hemiparesis | C3–5 | posterolateral | Surgery | Recovery |
| 27 | Female | Headache | C1-S1 | not reported | Conservative | Recovery |
| 79 | Male | Lower extremity paralysis | C2–4 | posterolateral | Surgery | Recovery |
Domenic- cci et al.,2007 [ 8] | 52 | Female | Hemiparesis | C4-T1 | posterolateral | Surgery | Recovery |
| 38 | Female | Upper extremity paralysis | C4 | posterolateral | Conservative | Recovery |
| 40 | Male | Upper extremity paralysis | C2-T2 | posterolateral | Surgery | Recovery |
| 44 | Female | Tetraplegia | C1–4 | anterior | Conservative | Recovery |
| 33 | Male | Tetraplegia | C4–7 | posterolateral | Surgery | Die |
| 38 | Male | Paraparesis | C6-T1 | anterior | Conservative | Recovery |
Present case | 55 | Male | Tetraplegia | C3-T3 | posterolateral | Surgery | Partial recovery |
Cervical spinal epidural hematoma is usually characterized by neck pain, scapular pain, and varying degrees of neurological deficits [
5]. An early MRI scan is necessary for this type of patient, and it can accurately determine the location and severity of the hematoma. Patients with mild neurological symptoms and a stable condition can be treated conservatively. In a case reported by Ryu et al. [
10], because the patient’s symptoms rapidly improved, no surgical treatment was required and he was discharged in only 1 week. Surgical treatment should be performed in patients with severe neurological deficits or progressive severe symptoms. In a case reported by Ling et al. [
7], surgery was performed after the patient was admitted for tetraplegia after neck manipulation. These authors believed an earlier surgical intervention would have delivered a better outcome and improvement. Our patient who had a severe nerve defect underwent surgical treatment and achieved good results. Surgery prevented further compression and edema of the cervical spinal cord, which created a favorable environment for subsequent recovery of nervous function.
How to improve the safety of cervical spine manipulation? It is important that every potential serious adverse event caused by vascular or other pathologies should be prevented. Thus, thorough patient interviewing, clinical assessment, interpretation and analysis are significant components needed to define an indication for cervical spine manipulation [
21]. Table
3 presents a summary of contraindications and precautions for cervical spine manipulation [
21]. Cervical spine manipulation should not be performed when contraindications are present [
17]. Prior to manipulation, a risk-benefit analysis should be performed and that includes the following three steps [
21] :①identifying a possible vasculogenic contribution or other serious pathology; ②determining whether there is an indication or contraindication for mobilization or manipulation; ③sessing the presence of any potential risk factors associated with potential serious adverse events which are reported to occur after cervical spine mobilization and/or manipulation. Potential risk factors, risk signals and contraindications can be found in interviews with patients and this information can provide a basis to create initial hypotheses to be further investigated in the clinical examination [
22]. Physical examination before manipulation is also necessary, because the examination of abnormal sensory and muscular strength of limbs maybe occur in patients with cervical epidural hematoma and a positive test can be regarded as an indicator of the patient’s risk of getting severe complications during a cervical manipulation. Such as spinal epidural hematoma can present with features ranging from simple pain with radiculopathy to complete paraplegia or quadriplegia [
23]. If we just adopt spinal manipulation because of stiffness and pain in the neck and ignore the abnormal results of other physical examinations, it may lead to serious consequences. The upper cervical spine instability tests and premanipulative vertebrobasilar insufficiency tests these tests can be valuable in detecting upper cervical spine instability or vertebrobasilar insufficiency, but their applicability as primary screening test has yet to be confirmed [
24,
25]. Moreover, cervical manipulation should not be performed at the end of range of cervical movement, particularly extension and rotation [
22].
Table 3
Contraindications and precautions to perform cervical spinal manipulation [
21]
(Acute) fracture | Inflammatory disease |
Relevant recent trauma | Rheumatoid arthritis |
Dislocation | Ankylosing spondylitis |
Ligamentous rupture | History of cancer |
Instability | Long-term steroid use |
Active cancer | Osteoporosis |
Acute myelopathy | Systemically unwell |
Spinal cord damage | Hypermobility syndromes |
Upper motor neuron lesions | Connective tissue disease |
Multi-level nerve root pathology | A first sudden episode before age 18 or after age 55 |
Worsening neurological function | Cervical anomalies |
Recent surgery | Local infection |
Acute soft tissue injury | Throat infection |
Unremitting, severe, non-mechanical pain | Recent manipulation by another health professional |
Unremitting night pain | Vascular disease |
Vertebral / carotid artery abnormalities | Blood clotting disorders / alterations in blood properties |
Vertebrobasilar insufficiency | Anticoagulant therapy |
Absence of a plausible mechanical explanation for the patient’s symptoms |
Immediately post-partum |
In conclusion, neck pain is common and recurrent in the general population, but in the absence of neurological signs and symptoms, there is no practical, clinically valid screening tests to identify underlying risks in patients with neck pain. So, history taking and patient characteristics are very important. Patients with a suspected hematoma should first be examined by MRI to make a definite diagnosis and guide further treatment.