Pooling of Csf in Frontal Brain Peer Review

Due southpontaneous intracranial hypotension has go a well-recognized and increasingly diagnosed crusade of headaches, especially in young and middle-anile adults.^9,11 When brain MRI findings and clinical presentation are typical of spontaneous intracranial hypotension, then treatment with epidural blood patching can continue without the need for further imaging. In patients with normal encephalon MRI findings and in those with persistent symptoms despite epidural blood patching, spinal imaging often is recommended to search for an underlying spinal CSF leak. Although in nearly patients a spinal CSF leak can be found, in some patients with spontaneous intracranial hypotension, no CSF leak can ever be demonstrated despite extensive evaluation with various imaging modalities such as MRI, radionuclide cisternography, CT myelography, T2-weighted MRI myelography, and intrathecal Gd-enhanced MRI myelography.^{1–3,eight–12,fifteen,17} This failure to localize a CSF leak can limit treatment options.

We recently reported the fortuitous discovery of CSF-venous fistulas in some patients with spontaneous intracranial hypotension.¹³ These fistulas were institute not only in patients with spinal CSF leaks but also in patients without any detectable CSF leak. We now report on the yield of finding a CSF-venous fistula past performing digital subtraction myelography (DSM) in patients with spontaneous intracranial hypotension but no CSF leak identifiable on conventional spinal imaging (i.eastward., non-DSM).

Methods

This report was approved by the Cedars-Sinai Medical Center institutional review board.

The patient population consisted of a group of 53 consecutive patients with spontaneous intracranial hypotension who underwent DSM between Apr 1, 2013, and September 30, 2014, but had no spinal CSF leak identifiable (i.e., presence of extradural CSF) on conventional CT myelography, MRI, or MR myelography. The diagnosis of spontaneous intracranial hypotension was fabricated using the criteria of the International Classification of Headache Disorders, Third Edition (ICHD-3)^iv (Tabular array ane).

Tabular array 1.

ICHD-3 diagnostic criteria for headache caused past spontaneous intracranial hypotension

Benchmark	Description
A	Whatever headache fulfilling criterion C
B	Low CSF pressure (<sixty mm CSF) &/or prove of CSF leakage on imaging
C	Headache developed in temporal relation to depression CSF force per unit area or CSF leakage or led to its discovery
D	Not ameliorate deemed for by some other ICHD-3 diagnosis

In all patients, the DSM technique, as described by Hoxworth et al.,^v,six,12 was used with some minor modifications. Briefly, DSM is performed with the patient in a country of general endotracheal anesthesia with deep paralysis and suspended respiration for maximal detail and temporal resolution. The patient is positioned prone in a biplane angiography suite with tilt-table capability. Cream padding or pillows are placed to reduce lumbar lordosis and overcome thoracic kyphosis. A fluoroscopically guided lumbar puncture is performed at the L2–3 level with a xx-guess spinal needle. An opening force per unit area is obtained at this fourth dimension (with the patient prone and in a state of general endotracheal anesthesia). Accurate needle positioning is confirmed with an injection of 0.5 ml of Omnipaque. The patient is then repositioned, based on the area of involvement, and the table is tilted to attain contrast menstruum to the cervicothoracic spine. Finally, contrast (ane ml/second) is injected manually while the patient's respiration is suspended for 40–60 seconds, and biplane subtraction images are acquired at 2 frames/second. Venous force per unit area is non measured.

Each patient completed a Migraine Inability Assessment (MiDAS) questionnaire to assess the severity of the symptoms before and later treatment.¹⁶ A MiDAS score of 0–v (Grade I) is considered equal to picayune or no inability, scores of 6–10 (Grade II) indicate mild disability, scores of 11–20 (Grade III) indicate moderate disability, and scores higher than 20 (Grade IV) indicate astringent disability.

For statistical analysis, ANOVA and the Fisher verbal test were used to compare groups and patient characteristics, respectively.

Results

Characteristics of the 53 patients are presented in Tabular array 2. The mean age of the 33 women and 20 men was 53.iv years (range 29–71 years). Every patient had presented with orthostatic headache. The duration of symptoms ranged from two to 172 months (mean 47 months). Every patient had undergone brain MRI exam, which in 42 (79%) of the patients showed 1 or more than of the typical stigmata (brain sagging, pachymeningeal enhancement, and subdural fluid collections) of spontaneous intracranial hypotension, although brain MRI findings had normalized in ix of these patients by the time of DSM. Although no CSF leak was identified on recent conventional spinal imaging in any of the 53 patients, ten patients had undergone spinal imaging between 2 and 9 years earlier that had shown the presence of a CSF leak. Eight patients had ever had normal brain MRI findings and never had a CSF leak revealed by spinal imaging, but all the patients had an abnormally depression opening pressure on lumbar puncture (less than six cm H_twoO). Thus, each patient showed objective evidence of spontaneous intracranial hypotension, as is required to fulfill ICHD-3 criteria.

TABLE two.

Characteristics of 53 patients with spontaneous intracranial hypotension and no CSF leak identifiable on conventional spinal imaging who underwent DSM

Variable	All Patients	Fistula		p Value
		Yes	No
No. (%) of patients	53	10 (18.9)	43 (81.i)
Age at diagnosis (yrs)				0.53
Mean (SD)	53.iv (ten.seven)	51.8 (9.vi)	54.3 (11.0)
Median (IQR)	54 (61–47)	53 (60–48)	55 (62–47)
Sexual activity (n [%])				0.0697
Female person	33 (62.3)	9 (90.0)	24 (55.8)
Male	20 (37.vii)	1 (10.0)	19 (44.2)
Duration of symptoms (mos)				0.38
Mean (SD)	47.0 (43.7)	37.6 (39.5)	49.2 (44.8)
Median (IQR)	37 (63–xv)	19 (57–eleven)	41 (68–18)
Low opening pressure (<half-dozen cm H2O) (due north [%])
At fourth dimension of initial lumbar puncture	25 (47.2)	vi (60.0)	nineteen (44.2)	0.49
At time of DSM	9 (17.0)	one (ten.0)	eight (18.6)	1.00
Positive MRI findings (n [%])
Baseline	42 (79.3)	eight (80.0)	34 (79.1)	ane.00
At time of DSM	33 (62.3)	half dozen (60.0)	27 (62.8)	one.00
Normalization of MRI findings at time of DSM (n [%])	ix (17.0)	2 (20.0)	7 (16.3)	1.00
CSF leak found on previous spinal imaging (north [%])				one.00
Positive	10 (18.9)	two (20.0)	8 (18.6)
Negative	43 (81.1)	viii (lxxx.0)	35 (81.4)

Each patient had undergone at least two epidural blood or fibrin-glue patch procedures, and 6 patients had undergone surgical handling of spinal CSF leak or spinal meningeal diverticula.

A CSF-venous fistula was found in 10 (xix%) of the 53 patients. The mean historic period of these 9 women and 1 man was 51.8 years (range 34–64 years). The elapsing of symptoms ranged from 5 to 121 months (mean 38 months). We were not able to place whatever factors associated with an increased likelihood of finding a CSF-venous fistula on DSM (Table 2). CSF-venous fistulas were constitute in ix (27%) of the 33 women and in i (5%) of the 20 men, but this event was of simply borderline statistical significance (p = 0.0697).

Every CSF-venous fistula was located in the thoracic spine, as shown in Fig. 1. Multiple fistulas were non encountered. The diameters of the venous channel were approximately i–two mm. Typical examples of these CSF-venous fistulas demonstrated by DSM are shown in Fig. 2 and Videos 1 and 2.

FIG. i.

Frequency of CSF-venous fistulas co-ordinate to spinal level. The view is from the back equally it is on DSM, and the right side is on the right. Figure is available in color online merely.

FIG. 2.

A–D: CSF-venous fistulas in patients with spontaneous intracranial hypotension: DSM images (frontal projection) showing contrast filling spinal veins (arrows). Effigy is available in color online only.

VIDEO i. DSM showing a CSF-venous fistula in a 39-year-old man at the T10–11 level on the left (compare with Fig. 2A). Copyright Wouter I. Schievink. Published with permission. Click here to view.

VIDEO two. DSM showing a CSF-venous fistula at the right T9–ten level in a 53-yr-old woman (compare with Fig. 2C). Copyright Wouter I. Schievink. Published with permission. Click here to view.

At the time of DSM, opening pressure was unmeasurably depression in ane patient and normal in nine patients (range 9–13 cm H_iiO). Opening pressure at the time of the initial lumbar puncture, all the same, was beneath normal in half-dozen patients (range 0–5.5 cm H_iiO).

Earlier treatment of the spinal CSF-venous fistula, the MiDAS class was 3 (moderate disability) in two patients and IV (severe disability) in 8 patients.

Four patients were treated initially with percutaneous fibrin sealant injection directed at the site of the CSF-venous fistula. In that location were no complications from the percutaneous fibrin sealant injection. The injection resulted in complete resolution of symptoms in 1 patient (MiDAS Grade I) (follow-up 6 months), whereas symptoms persisted in the other 3 patients. These 3 patients, as well as the other vi patients, underwent surgery for their fistula. In 6 patients, the fistula consisted of a network of engorged veins surrounding the proximal spinal nerve root sleeve (with or without cyst development). Treatment consisted of bipolar coagulation of this venous network. In three patients, the fistula consisted of a single discrete draining venous channel (Fig. 3) near the origin of, simply not involving, the spinal nerve root sleeve. Handling consisted of clipping and ligation of the venous aqueduct (Video iii) in 2 patients and bipolar coagulation of the venous channel in one patient.

FIG. iii.

Intraoperative photographs showing the anatomy of a CSF-venous fistula. Upper: The single venous channel is attached to the lateral common thecal sac (pointer). Lower: The fistula is treated by prune ligation. Figure is available in color online only.

VIDEO 3. Unedited movie clip of surgical treatment of a single-channel CSF-venous fistula at the right T11–12 level in a 62-year-old adult female (compare with Fig. 2B). Copyright Wouter I. Schievink. Published with permission. Click here to view.

In that location were no complications from the microsurgical handling. Surgery resulted in complete resolution of symptoms in eight patients (MiDAS Form I; follow-up vii–23 months), whereas in 1 patient, symptoms recurred after 4 months. Repeat DSM did not evidence the CSF-venous fistula, and treatment with multiple epidural blood patches resumed. The stigmata of spontaneous intracranial hypotension on encephalon MRI resolved in all 6 patients.

Discussion

In this written report, we plant spinal CSF-venous fistulas in approximately one-fifth of the patients with spontaneous intracranial hypotension merely no CSF leak identifiable on conventional spinal imaging. Merely single CSF-venous fistulas were found, and just in the thoracic spine. Treatment of these fistulas was uncomplicated, usually consisted of surgery, and was successful in 90% of the patients. Symptom relief was durable, but follow-upwardly has been limited because of the very recent discovery of these fistulas. At surgery, the fistula consisted of either a network of veins surrounding the spinal nervus root sleeve or a single draining venous channel almost the origin of the spinal nerve root sleeve. It is not clear if the fistulas stand for aberrant anatomical structures or abnormally dilated vascular conduits. Arachnoid granulations along the proximal spinal nervus root sleeves extending into the lumen of a vein have been described,^{half dozen} and intradural and extradural venous engorgement is a common finding in patients with spontaneous intracranial hypotension.⁷

Spinal CSF-venous fistulas were plant in patients with normal brain MRI findings and in those with abnormal encephalon MRI findings and in patients with previous spinal imaging showing a CSF leak and in those without such a history. These fistulas were found more commonly in women than in men, but this finding did non reach statistical significance.

Information technology is not surprising that these CSF-venous fistulas were non detected by cantankerous-sectional imaging techniques (e.g., MRI or CT myelography, or even conventional myelography). Although not as fast every bit an arteriovenous fistula, this entity requires rapid imaging with superior spatial and temporal resolution, which is best achieved with DSM.

The inability to find a spinal CSF leak limits treatment options for patients with intractable spontaneous intracranial hypotension, and we suggest that DSM exist considered for this patient population, although the actual yield of finding a CSF-venous fistula is relatively low.

The underlying cause of spontaneous intracranial hypotension in those with normal results from spinal imaging, including DSM, remains speculative and requires farther investigation. A CSF leak below the level of detection of electric current imaging engineering science, an intermittent or very dull CSF leak, unusually rapid assimilation of spinal CSF, and pooling of CSF are all potential mechanisms underlying spontaneous intracranial hypotension with patients with normal spinal imaging results. However, studies to rule out a CSF leak at the level of the skull base practise non demand to be performed, because spontaneous intracranial hypotension is not caused by cranial CSF leaks.¹⁴

Conclusions

In this report, we found CSF-venous fistulas in approximately one-fifth of the patients with recalcitrant spontaneous intracranial hypotension but no CSF leak identifiable on conventional spinal imaging. We suggest that DSM exist considered in this patient population.

References

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Disclosures

The authors study no disharmonize of interest concerning the materials or methods used in this report or the findings specified in this paper.

Author Contributions

Conception and design: Schievink, Moser, Maya. Conquering of data: all authors. Analysis and interpretation of data: all authors. Drafting the commodity: Schievink. Critically revising the article: Schievink. Reviewed submitted version of manuscript: Schievink. Canonical the final version of the manuscript on behalf of all authors: Schievink. Statistical analysis: Schievink.

Supplemental Data

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Source: https://thejns.org/spine/view/journals/j-neurosurg-spine/24/6/article-p960.xml

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