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Froin Syndrome After Spinal Cord Injury
Case Report
Froin Syndrome After Spinal Cord Injury Thomas Decramer1, Anke Wouters2, Carlotte Kiekens3, Tom Theys1
Key words Froin syndrome - Intrathecal drug therapy - Spinal cord injury - Xanthochromia
- BACKGROUND:
Abbreviations and Acronyms CSF: Cerebrospinal fluid MRI: Magnetic resonance imaging SCI: Spinal cord injury
- CASE
-
From the Departments of 1Neurosurgery, 2Neurology, and 3 Physical and Rehabilitation Medicine, University Hospitals, Leuven, Belgium To whom correspondence should be addressed: Thomas Decramer, M.D. [E-mail: [email protected]]
Froin syndrome is characterized by xanthochromia and hypercoagulability of the cerebrospinal fluid (CSF) due to elevated protein levels. This entity results from blockage of the spinal canal by a mass lesion leading to an isolated caudal CSF space.
DESCRIPTION: A 48-year-old male, who developed spasticity after a C6 spinal cord injury (SCI) 20 years earlier, presented with subobstruction of his intrathecal baclofen pump. A catheter access port aspiration revealed an extremely high protein concentration (38 g/L) with no signs of infection. Froin syndrome was confirmed when magnetic resonance imaging showed a complete obstruction of the spinal canal at the SCI level.
- CONCLUSIONS:
We report the first case of Froin syndrome after SCI. Froin syndrome can impact intrathecal drug delivery and CSF diagnostics.
Citation: World Neurosurg. (2019) 127:490-491. https://doi.org/10.1016/j.wneu.2019.04.198 Journal homepage: www.journals.elsevier.com/worldneurosurgery
refractory to increasing intrathecal baclofen dosages.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
BACKGROUND A 48-year-old male developed severe spasticity after a C6 spinal cord injury (SCI; American Spinal Injury Association A) due to a motor vehicle accident 20 years earlier. Three months after an initial good effect from intrathecal baclofen therapy, he presented with increased spasticity
CASE DESCRIPTION A catheter-related problem was suspected and a catheter access port aspiration revealed highly viscous cerebrospinal fluid (CSF) with xanthochromia (Figure 1A). CSF analysis revealed an extremely high protein concentration (38 g/L), a normal glucose (73 mg/dL) and few red blood cells. The patient did not show any meningeal signs and CSF cultures were negative. Spasticity improved for 2 weeks
Figure 1. (A) Macroscopic appearance of the cerebrospinal fluid (CSF). (B) Cervical magnetic resonance imaging (MRI), T2-weighted imaging demonstrating an obstruction of the spinal canal (arrow) due to spinal cord injury with scarring and cyst
490
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but then recurred. A new attempt to perform a catheter access port aspiration was unsuccessful indicating a complete catheter obstruction. Additional magnetic resonance imaging (MRI) of the spine revealed an obstruction of the spinal canal at the site of SCI (Figure 1B) with the CSF below being slightly more hyperintense. At the lumbar level, the caudal part of the thecal sac was filled with debris (Figure 1C). In 1903, Georges Froin described extremely high CSF protein levels (>5 g/L) causing xanthochromia and hypercoagulability.1 Froin syndrome results from a
formation in the cervical cord. The CSF compartment caudal to the obstruction is slightly more hyperintense. (C) Lumbar MRI, T2-weighted imaging shows that the caudal part of the thecal sac is filled with debris (arrow).
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.04.198
CASE REPORT THOMAS DECRAMER ET AL.
blockage of the spinal canal, causing an isolated CSF space caudal to the obstruction, which has an elevated protein level due to venous congestion or inflammation by which blood plasma products enter the subarachnoid space.2 Froin syndrome is typically caused by a neoplasm or inflammation obstructing normal CSF flow.3-5 CONCLUSION To our knowledge, Froin syndrome was never reported after SCI. Knowledge of this syndrome is important as it may impact intrathecal therapy due to increased viscosity and hypercoagulability
FROIN SYNDROME AFTER SPINAL CORD INJURY
of CSF and thus cause catheter (sub)obstruction. At lumbar puncture or catheter access port aspiration, the CSF aspect can be misdiagnosed as a hemorrhage or infection.
4. Kwon SK, Kim MW. Pseudo-Froin’s syndrome, xanthochromia with high protein level of cerebrospinal fluid. Kor J Anesthesiol. 2014;67:S58-S59.
5. Dancel R, Shaban M. Images in clinical medicine. Froin’s syndrome. N Engl J Med. 2016;374:1076.
REFERENCES 1. Froin G. Inflammations méningées avec chromatique, fibrineuse et cytologique du liquide céphalorachidien. Gazette Hôpitaux. 1903;76:1005-1006. 2. Greenfield JG. Original papers: on Froin’s syndrome, and its relation to allied conditions in the cerebrospinal fluid. J Neurol Psychopathol. 1921;2: 105-141. 3. Mirza S, Adams WM, Corkhill RA. Froin’s syndrome revisited, 100 years on. Pseudo-Froin’s syndrome on MRI. Clin Radiol. 2008;63:600-604.
WORLD NEUROSURGERY 127: 490-491, JULY 2019
Conflict of interest statement: Tom Theys is a Senior Clinical Investigator of FWO Flanders (FWO 1830717N). Received 9 April 2019; accepted 22 April 2019 Citation: World Neurosurg. (2019) 127:490-491. https://doi.org/10.1016/j.wneu.2019.04.198 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
www.journals.elsevier.com/world-neurosurgery
491
Froin Syndrome After Spinal Cord Injury Thomas Decramer1, Anke Wouters2, Carlotte Kiekens3, Tom Theys1
Key words Froin syndrome - Intrathecal drug therapy - Spinal cord injury - Xanthochromia
- BACKGROUND:
Abbreviations and Acronyms CSF: Cerebrospinal fluid MRI: Magnetic resonance imaging SCI: Spinal cord injury
- CASE
-
From the Departments of 1Neurosurgery, 2Neurology, and 3 Physical and Rehabilitation Medicine, University Hospitals, Leuven, Belgium To whom correspondence should be addressed: Thomas Decramer, M.D. [E-mail: [email protected]]
Froin syndrome is characterized by xanthochromia and hypercoagulability of the cerebrospinal fluid (CSF) due to elevated protein levels. This entity results from blockage of the spinal canal by a mass lesion leading to an isolated caudal CSF space.
DESCRIPTION: A 48-year-old male, who developed spasticity after a C6 spinal cord injury (SCI) 20 years earlier, presented with subobstruction of his intrathecal baclofen pump. A catheter access port aspiration revealed an extremely high protein concentration (38 g/L) with no signs of infection. Froin syndrome was confirmed when magnetic resonance imaging showed a complete obstruction of the spinal canal at the SCI level.
- CONCLUSIONS:
We report the first case of Froin syndrome after SCI. Froin syndrome can impact intrathecal drug delivery and CSF diagnostics.
Citation: World Neurosurg. (2019) 127:490-491. https://doi.org/10.1016/j.wneu.2019.04.198 Journal homepage: www.journals.elsevier.com/worldneurosurgery
refractory to increasing intrathecal baclofen dosages.
Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
BACKGROUND A 48-year-old male developed severe spasticity after a C6 spinal cord injury (SCI; American Spinal Injury Association A) due to a motor vehicle accident 20 years earlier. Three months after an initial good effect from intrathecal baclofen therapy, he presented with increased spasticity
CASE DESCRIPTION A catheter-related problem was suspected and a catheter access port aspiration revealed highly viscous cerebrospinal fluid (CSF) with xanthochromia (Figure 1A). CSF analysis revealed an extremely high protein concentration (38 g/L), a normal glucose (73 mg/dL) and few red blood cells. The patient did not show any meningeal signs and CSF cultures were negative. Spasticity improved for 2 weeks
Figure 1. (A) Macroscopic appearance of the cerebrospinal fluid (CSF). (B) Cervical magnetic resonance imaging (MRI), T2-weighted imaging demonstrating an obstruction of the spinal canal (arrow) due to spinal cord injury with scarring and cyst
490
www.SCIENCEDIRECT.com
but then recurred. A new attempt to perform a catheter access port aspiration was unsuccessful indicating a complete catheter obstruction. Additional magnetic resonance imaging (MRI) of the spine revealed an obstruction of the spinal canal at the site of SCI (Figure 1B) with the CSF below being slightly more hyperintense. At the lumbar level, the caudal part of the thecal sac was filled with debris (Figure 1C). In 1903, Georges Froin described extremely high CSF protein levels (>5 g/L) causing xanthochromia and hypercoagulability.1 Froin syndrome results from a
formation in the cervical cord. The CSF compartment caudal to the obstruction is slightly more hyperintense. (C) Lumbar MRI, T2-weighted imaging shows that the caudal part of the thecal sac is filled with debris (arrow).
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.04.198
CASE REPORT THOMAS DECRAMER ET AL.
blockage of the spinal canal, causing an isolated CSF space caudal to the obstruction, which has an elevated protein level due to venous congestion or inflammation by which blood plasma products enter the subarachnoid space.2 Froin syndrome is typically caused by a neoplasm or inflammation obstructing normal CSF flow.3-5 CONCLUSION To our knowledge, Froin syndrome was never reported after SCI. Knowledge of this syndrome is important as it may impact intrathecal therapy due to increased viscosity and hypercoagulability
FROIN SYNDROME AFTER SPINAL CORD INJURY
of CSF and thus cause catheter (sub)obstruction. At lumbar puncture or catheter access port aspiration, the CSF aspect can be misdiagnosed as a hemorrhage or infection.
4. Kwon SK, Kim MW. Pseudo-Froin’s syndrome, xanthochromia with high protein level of cerebrospinal fluid. Kor J Anesthesiol. 2014;67:S58-S59.
5. Dancel R, Shaban M. Images in clinical medicine. Froin’s syndrome. N Engl J Med. 2016;374:1076.
REFERENCES 1. Froin G. Inflammations méningées avec chromatique, fibrineuse et cytologique du liquide céphalorachidien. Gazette Hôpitaux. 1903;76:1005-1006. 2. Greenfield JG. Original papers: on Froin’s syndrome, and its relation to allied conditions in the cerebrospinal fluid. J Neurol Psychopathol. 1921;2: 105-141. 3. Mirza S, Adams WM, Corkhill RA. Froin’s syndrome revisited, 100 years on. Pseudo-Froin’s syndrome on MRI. Clin Radiol. 2008;63:600-604.
WORLD NEUROSURGERY 127: 490-491, JULY 2019
Conflict of interest statement: Tom Theys is a Senior Clinical Investigator of FWO Flanders (FWO 1830717N). Received 9 April 2019; accepted 22 April 2019 Citation: World Neurosurg. (2019) 127:490-491. https://doi.org/10.1016/j.wneu.2019.04.198 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
www.journals.elsevier.com/world-neurosurgery
491