Acute Subdural Hemorrhage as the Initial Presentation of Intracranial Hypotension Following Cervical Chiropractic Manipulation: A Case Report and Systematic Review

Abstract

Spontaneous intracranial hypotension (SIH) still remains an underdiagnosed etiology of new-onset headache. Important risk factors include chiropractic manipulation (CM). We present a case of a 36-year-old Filipino woman who presented with severe bifrontal and postural headache associated with dizziness, vomiting, and doubling of vision. A cranial computed tomography scan was done which showed an acute subdural hematoma (SDH) at the interhemispheric area. Pain medications were given which afforded minimal relief. On history, the headaches occurred 2 weeks after cervical CM. Cranial and cervical magnetic resonance imaging revealed findings supportive of intracranial hypotension and neck trauma, respectively. The patient improved with conservative management. We found 12 articles on SIH and CM after a systematic review of literature. Eleven patients (90.9%) initially presented with orthostatic headache. Eight patients (66.7%) were initially treated conservatively but only 5 (62.5%) had complete recovery. Recovery was achieved within 14 days from start of supportive therapy. Among the 3 patients who failed conservative treatment, 2 underwent non-directed epidural blood patch and one required neurosurgical intervention. This report highlights that a thorough history is warranted in patients with new onset headache. A history of CM must be actively sought. The limited evidence from the case reports showed that patients with SIH and SDH but with normal neurologic examination and minor spinal pathology can be managed conservatively for less than 2 weeks. This review showed that conservative treatment in a closely monitored environment may be an appropriate first line treatment.

Introduction

Subdural hemorrhages (SDH) are often caused by head injury. Other common etiologies include ruptured aneurysms or arteriovenous malformations, intracranial tumoral bleed, and coagulopathy. ¹ It can also be from a neglected minor head trauma, especially in the elderly population or from a spontaneous intracranial hypotension (SIH) resulting from a cerebrospinal fluid (CSF) leak. ¹ An underdiagnosed etiology is SIH following chiropractic manipulation (CM). ² We describe a female who presented with an acute SDH as the initial manifestation of SIH after CM. There is limited and conflicting publications on the treatment options and outcomes of SIH following CM, hence we performed a systematic review of literature on SIH secondary to CM.

Case Description and Systematic Review

A healthy 36-year-old Filipino female presented with severe bifrontal and postural headache associated with dizziness, vomiting and doubling of vision. A cranial computed tomography (CT) scan was done which showed an acute SDH at the interhemispheric area (Figure 1A). Pain medications were given which afforded minimal relief. There was no history of head trauma, blood dyscrasias or blood thinner intake. The patient noted that she experienced daily persistent headaches 2 weeks after axial tension and rotatory manipulation of her neck. During the CM, she immediately felt sharp pain on her neck. Continuous neck stiffness was felt post-manipulation. Her current neurologic examination was normal.

Figure 1.

Cranial and spinal imaging. (A) The axial plain cranial CT on admission revealed an acute subdural hemorrhage along the falx cerebri. (B) The axial cranial MRI showed thickening of the dura and diffuse and symmetric enhancement. (C) The sagittal cervical MRI revealed a soft-tissue hyperintensity (STIR sequence) in the interspinous C1-C2 level. (D) The axial slices showed epidural fluid collection at the C2- C3 levels (white arrow). Follow up imaging after 2 months (E) Axial plain CT showing resolution of the subdural hemorrhage and (F) Sag STIR cervical MRI done after 3 months showed resolution of the hyperintense signal in the interspinous region at C1-C2.

Cranial magnetic resonance imaging (MRI) and angiography revealed diffuse pachymeningeal enhancement, slit-like lateral ventricles, prominent venous sinuses (Figure 1B), and a mamillo-pontine distance of 5.1 mm (normal value: >5.5 mm) suggestive of intracranial hypotension. There were no signs of carotid or vertebral artery dissection. The cervical MRI revealed a soft tissue hyperintensity at the C1-C2 interspinous area on short tau inversion recovery (STIR) sequence (Figure 1C). There was also note of an epidural fluid collections at the C2- C3 (Figure 1D) and C3-C4 levels which was considered as the area of the CSF leak. The MRI of the rest of the spine was unremarkable.

Conservative management for 12 days was done which consisted of bed rest, analgesics, intravenous hydration and oral fluids. A follow up plain cranial CT scan after 2 months showed resolution of the SDH along the posterior falx (Figure 1E). Cervical MRI after 3 months also showed resolution of the C1-C2 STIR soft-tissue hyperintensity (Figure 1F). The patient had no recurrence of headache on follow-up 3 months after.

We conducted a systematic review of studies which associated SIH with CM. We excluded cases secondary to post lumbar puncture or spine surgery, recent trauma other than those following CM, cases secondary to connective tissue disorders, or otherwise those who were reported as spontaneous. We searched the following electronic databases until May 15, 2020 for relevant articles: PubMed by MEDLINE, CENTRAL by The Cochrane Library, Scopus and LILACS. The general search terms used were “intracranial hypotension”, “chiropractic manipulation”, “orthostatic headache” and “low cerebrospinal fluid pressure”. All titles and abstracts of the records retrieved from the systematic search were screened. All articles were obtained in full text. We included all articles in English which fulfilled the eligibility criteria (Figure 2). Duplicates were removed. Three full text articles were excluded because they were not in English. The following data were collected: age, sex, spinal manipulation technique, time interval from spinal manipulation to symptom onset, clinical presentation, opening pressure during lumbar puncture, cranial and spinal imaging, CSF localization technique, management, and recovery. Descriptive statistics were used to tabulate the data.

Figure 2.

Preferred reporting items for systematic reviews and meta-analyses flow diagram.

Results

Out of 190 records retrieved from the systematic search, 12 articles were included. (Table 1). Including the present case, information from a total of 13 patients were collated. The mean age was 41.6 ± 7.8 years [range, 29 – 54] and ten patients (83.3%) were female. All patients presented with orthostatic headache^2-13 while 5 patients had vomiting (41.7%).^4-6,11,12

Table 1.

Cases With Intracranial Hypotension Following Chiropractic Manipulation.

	Age/sex	SMT technique	Interval after SMT	Clinical presentation	Opening pressure (LP)	Cranial imaging findings	Spinal MRI findings	CSFleak detection procedure	Management/ Outcome
Chung et al., 2000 2	44/ F	Not described	Not described	Orthostatic headache, neck stiffness	Not mentioned	Not mentioned	Not mentioned	RC: no CSF leak	Supportive/ Complete
Jeret et al., 2001 3	34/ M	Rapid rotatory neck manipulation	36 hours	Orthostatic headache, nape pain, dizziness	80 mm H20	Plain CT: normal MRI + C: normal	None	Myelography (after 5 weeks): no CSF leak	Epidural blood patch x 1/ Partial
Beck et al., 2003 4	40/ F	Axial tension and rotation	Within 24 hours	Orthostatic headache, nausea and vomiting, doubling of vision	Not mentioned	MRI + C: diffuse PE MRI: bilateral subdural effusion	Ax T2 Cervical: CSF accumulation in the dorsal perivertebral space around the dural sac at C1-C2 level	None	Supportive/ Complete
Suh et al., 2005 5	37/ F	Axial tension and rotation	1 day	Orthostatic headache, nausea and vomiting, nape pain	40 mm H20	CT + C: diffuse PE MRI + C: PE	Sag T2 Thoracic: epidural and subdural fluid collection Sag + Thoracic and Lumbar + C: diffuse dural enhancement and epidural venous engorgement	None	Initially supportive then epidural blood patch x 1, 20ml/ Complete
Straus et al., 2005 6	54/ F	Axial tension and rotation	Immediate	Orthostatic headache, vomiting	30 mm H20	MRI: bilateral PE	None	RC: CSF leak upper thoracic level	Initially supportive, then epidural blood patch x 1, 20ml/ Complete
Matthews et al., 2006 7	51/ F	Not described	7 days	Headache, diplopia	No LP	MRI: bilateral PE	None	CT Myelography: CSF leak at C2	epidural blood patch x 1/ Complete
Morelli et al., 2006 8	49/ M	Rotation and hyperexten-sion	1 day	Orthostatic headache, tinnitus	No LP	Plain CT: normal MRI + C: Fronto-parietal PE	MR whole spine + myelopgraphy	MR myelography: ararachnoid cyst of of 3rd cervical root + CSF leakage	Supportive/ Complete
Prasad et al., 2006 9	37/ F	Axial tension and rotation	Immediate	Orthostatic headache	Not mentioned	Plain CT: bilateral subdural hygroma; tonsillar herniation	Cervical: extra-axial fluid collection	Planar scintigraphy: abnormal radiotracer activity at the anterior thoracolumbar spine	epidural blood patch x1/ Complete
Kurbanyan et al., 2007 10	46/ F	Axial tension and rotation	Not specified	Orthostatic headache, neck stiffness, diplopia	Not mentioned	None	Sag cervical + C: enhancement of the basilar meninges	None	Supportive/ Complete
Kusnezov et al., 2013 11	29/ F	Axial tension and rotation	1 week	Orthostatic headache, nausea and vomiting	None	None	Ax T2 Cervical: ventral C6 epidural fluid	None	Supportive/ Complete
Wilson et al., 2015 12	32/ F	Axial tension and lateral flexion	Not specified	Orthostatic headache, dizziness, vomiting, blurring of vision	None	MRI: flattening of the pons, cerebellar tonsil descent and PE	MRI: C5/6 central disc protrusion indenting the thecal sac and a ventral epidural CSF collection extending from C6 to T7	None	C5/6 anterior cervical discectomy; removal of calcified disk fragment; Surgicel was placed in C5/6 interspace; Solis polyether ether ketone (PEEK) was placed
Lin et al., 2016 13	36/ F	Tension on the occipital area, posterior nuchal area, and bilateral shoulders	1 day	Orthostatic headache	5 mm H20	MRI + C: brain sagging, faint leptomeningeal enhancement at the cerebral hemispheres and bilateral engorgement of the transverse sinuses	Spine MRI: periradicular CSF leaks, dural tear at the C7-T1 junction, marked CSF leakage near bilateral apical pleural regions	MRI myelography: symmetric SPFs permitted flow of CSF from the subarachnoid space into the pleural cavities over the bilateral lungs	targeted epidural blood patch x 3/ Complete
Fernando et al., 2021 (present case)	36/ F	Axial tension and rotation	2 weeks	Orthostatic headache, dizziness, nausea and vomiting, doubling of vision	None	Plain CT: subdural hemorrhage, interhemispheric MRI +C: diffuse PE; sagging brain signs	Sag STIR Cervical: hyperintense signal in the interspinous region at C1-C2 level	None	Supportive/ Complete

F = female, M = male, SMT = spinal manipulation therapy; LP = lumbar puncture; RC = radioisotope cisternography; MRI = magnetic resonance imaging; C = contrast; CT = computed tomography; PE = pachymeningeal enhancement; Sag = sagittal

Axial tension (n = 6, 50%)^4-6,9-11 and rotation (n = 8, 66.7%)^3-6,8-11 were the most common mechanisms of CM. The median time from CM to symptom onset was 24 hours [range, 0 – 168 hours]. However, not all studies specified the onset of symptoms after CM.^2,10,12 Only 8 patients had cranial MRI. The most common cranial imaging findings were diffuse pachymeningeal enhancement (n = 6, 75%),^4-9 subdural hygroma (n = 2, 25%),^2,8 tonsillar herniation (n = 1, 12.5%) ⁹ and venous sinus engorgement (n = 1, 12.5%). ¹³ A spine MRI was done in 7 patients (58.3%) which revealed extra-axial fluid collections at the epidural space, ventral to the thecal sac.^3,5,8-11,13 The number of affected levels were 2 [1 – 2]. Only 7 patients (58.3%) had CSF leak localization procedures such as myelography (n = 4)^3,7,8,13 or cisternography (n = 2).^2,6 Five of these 7 patients had CSF leak detected at the thoracic (n = 3),^5,9,13 cervical (n = 2)^7,8 and lumbar (n = 1) ⁹ levels.

Seven patients (58.3%) were initially treated conservatively,^{2,4,5,6,8,10,11} with 2 patients eventually receiving an epidural blood patch.^5,6 Four patients were immediately treated with an epidural blood patch (EBP).^3,7,9,13 Five patients (41.7%) recovered after supportive management.^2,4,8,10,11 The median time to complete recovery was achieved in 13 days [range, 12 – 14]. One patient was given an EBP after 18 days of failed supportive therapy. ⁵ Two patients who had non-directed EBPs had complete recovery after the first blood patch.^7,9 All cases had complete recovery after one to 3 EBPs.^3,7,9,13

One case was different from the 11 patients included in this review because it was the only case that presented with blurring of vision, central disc protrusion by spine MRI, and an epidural fluid collection spanning 9 vertebral levels. ¹² The treatment of this case was also different because an initial course of supportive management lasting for 28 days failed and complete recovery was only attained after surgery.

Discussion

Our case was unique from the other published reports because she presented with an acute interhemispheric SDH, headache onset 2 weeks after CM, and an area of soft-tissue injury at the interspinous C1-C2 level with a nearby epidural CSF collection on spine MRI. Despite having a structural pathology, our case did not follow the clinical course of the report of Wilson et al. ¹² Our patient had a complete recovery 12 days after supportive management – within the median recovery time of the series.

Medical management includes bed rest, hydration, analgesics, and caffeine. Caffeine can be given either by the intravenous route (500 mg in 500 mL saline over 2 hours repeated once or twice) or by simply increasing the oral intake of caffeinated beverages although the evidence for the latter is limited. ¹⁴ Conservative management is initially employed since leaks occasionally will resolve spontaneously. ¹⁵ Based on the current but limited data presented, supportive therapy may be continued for up to 2 weeks but the patient’s preference must be taken into account. If the patient remains symptomatic beyond 2 weeks, 1 to 3 non-directed EBPs may be given. If the patient is still symptomatic after these, a CSF localization procedure and a directed EBP can be done. ¹⁵ If these strategies are unsuccessful, surgical interventions may be necessary. ¹⁵

Data on the risk of CSF leak after CM is limited on the case reports reviewed. The data on the safety of CM is still insufficient. ¹¹ Mechanical disruption of the thecal sac with subsequent loss of CSF seems to be the main pathophysiological mechanism.^6,12 The fragility of the spinal dura at the level of the nerve root sleeves predisposes to the formation of meningeal diverticula following a traumatic event. ⁸ The commonly reported major complications CM were stroke accompanied by arterial dissection, phrenic nerve injury and myelopathy, and minor side effects such as stiffness, radiating pain, dizziness, and nausea. ¹¹ Accurate reporting of adverse events are also lacking. SIH after CM has rarely been reported, as seen on our systematic review.

This case report and literature review has several limitations. Not all studies specified the onset of symptoms after CM. There is no recommended standard work up. Our patient did not agree to a lumbar puncture. Only 8 studies reported cranial imaging and only 7 had spine MRI. A CSF localization procedure was not done in four reports including the present case. The time frames for supportive management were not indicated in other studies.

It is essential to note that a history of CM must be actively sought in patients presenting with SIH. A high level of clinical suspicion is necessary as misdiagnoses can cause significant delay and pain to these patients where supportive management is intuitive and successful. Our case highlights that a careful and thorough history is warranted in patients with new onset headache and acute SDH on imaging. The limited evidence from the case report shows that patients with SIH and SDH but with normal neurologic examination and minor spinal pathology may be managed conservatively for less than 2 weeks. This review has shown that conservative treatment in a closely monitored environment may be an appropriate first line treatment.

ORCID iD

Emmaline Z. Fernando, MD https://orcid.org/0000-0002-9345-871X

Roland Dominic G. Jamora, MD https://orcid.org/0000-0001-5317-7369