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Gowers' intrasyringeal hemorrhage associated with Chiari type I malformation in Noonan syndrome.

Mitsuhara T, Yamaguchi S, Takeda M, Kurisu K - Surg Neurol Int (2014)

Bottom Line: She presented progressive gait deterioration and acute urinary dysfunction, indicating conus medullaris syndrome.Initial magnetic resonance imaging revealed massive hemorrhage in the intrasyringeal cavity of the conus medullaris.The patient underwent surgical removal of the intrasyringeal hematoma and her neurological symptoms improved postoperatively.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurosurgery, Graduate School of Biomedical Sciences, Hiroshima University, 1-2-3 Kasumi, Minami.ku, Hiroshima 734-8551, Japan.

ABSTRACT

Background: Idiopathic hemorrhage in a syrinx is a rare entity known as Gowers' intrasyringeal hemorrhage. Bleeding confined to the syrinx cavity causes severe, sometimes acute, neurological deficits. We report a case of intrasyringeal hemorrhage into a preexisting lumbosacral syrinx associated with Chiari type I malformation.

Case description: A 39-year-old female with Noonan syndrome underwent foramen magnum decompression and a cervical syrinx-subarachnoid shunt for Chiari type I malformation-associated syringomyelia 7 years ago. She presented progressive gait deterioration and acute urinary dysfunction, indicating conus medullaris syndrome. Initial magnetic resonance imaging revealed massive hemorrhage in the intrasyringeal cavity of the conus medullaris. The patient underwent surgical removal of the intrasyringeal hematoma and her neurological symptoms improved postoperatively.

Conclusion: Although Gowers' intrasyringeal hemorrhage is rare, this entity should be taken into consideration in patients with syringomyelia showing acute neurological deterioration.

No MeSH data available.


Related in: MedlinePlus

MR images (a) T1-weighted image, (c- f) T2-weighted images) show a heterogeneous mass on the caudal end of the syringomyelic cavity suggesting hematomyelia. Partial gadolinium enhancement is revealed at the centre of the hematomyelia (b, arrowed)
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Figure 2: MR images (a) T1-weighted image, (c- f) T2-weighted images) show a heterogeneous mass on the caudal end of the syringomyelic cavity suggesting hematomyelia. Partial gadolinium enhancement is revealed at the centre of the hematomyelia (b, arrowed)

Mentions: On admission in 2010, she experienced gait disturbance and dysesthesia of both the legs and perineal region, which indicated conus medullaris syndrome. Routine hematological, serological, and blood coagulation examinations were all normal. Magnetic resonance (MR) imaging showed a large syringomyelic cavity extending from the C1 level to the conus. Both T1- and T2-weighted images showed a heterogeneous mass on the caudal end of the syringomyelic cavity, suggesting hematomyelia [Figure 2]. Partial gadolinium enhancement was revealed at the centre of the hematomyelia.


Gowers' intrasyringeal hemorrhage associated with Chiari type I malformation in Noonan syndrome.

Mitsuhara T, Yamaguchi S, Takeda M, Kurisu K - Surg Neurol Int (2014)

MR images (a) T1-weighted image, (c- f) T2-weighted images) show a heterogeneous mass on the caudal end of the syringomyelic cavity suggesting hematomyelia. Partial gadolinium enhancement is revealed at the centre of the hematomyelia (b, arrowed)
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3927095&req=5

Figure 2: MR images (a) T1-weighted image, (c- f) T2-weighted images) show a heterogeneous mass on the caudal end of the syringomyelic cavity suggesting hematomyelia. Partial gadolinium enhancement is revealed at the centre of the hematomyelia (b, arrowed)
Mentions: On admission in 2010, she experienced gait disturbance and dysesthesia of both the legs and perineal region, which indicated conus medullaris syndrome. Routine hematological, serological, and blood coagulation examinations were all normal. Magnetic resonance (MR) imaging showed a large syringomyelic cavity extending from the C1 level to the conus. Both T1- and T2-weighted images showed a heterogeneous mass on the caudal end of the syringomyelic cavity, suggesting hematomyelia [Figure 2]. Partial gadolinium enhancement was revealed at the centre of the hematomyelia.

Bottom Line: She presented progressive gait deterioration and acute urinary dysfunction, indicating conus medullaris syndrome.Initial magnetic resonance imaging revealed massive hemorrhage in the intrasyringeal cavity of the conus medullaris.The patient underwent surgical removal of the intrasyringeal hematoma and her neurological symptoms improved postoperatively.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurosurgery, Graduate School of Biomedical Sciences, Hiroshima University, 1-2-3 Kasumi, Minami.ku, Hiroshima 734-8551, Japan.

ABSTRACT

Background: Idiopathic hemorrhage in a syrinx is a rare entity known as Gowers' intrasyringeal hemorrhage. Bleeding confined to the syrinx cavity causes severe, sometimes acute, neurological deficits. We report a case of intrasyringeal hemorrhage into a preexisting lumbosacral syrinx associated with Chiari type I malformation.

Case description: A 39-year-old female with Noonan syndrome underwent foramen magnum decompression and a cervical syrinx-subarachnoid shunt for Chiari type I malformation-associated syringomyelia 7 years ago. She presented progressive gait deterioration and acute urinary dysfunction, indicating conus medullaris syndrome. Initial magnetic resonance imaging revealed massive hemorrhage in the intrasyringeal cavity of the conus medullaris. The patient underwent surgical removal of the intrasyringeal hematoma and her neurological symptoms improved postoperatively.

Conclusion: Although Gowers' intrasyringeal hemorrhage is rare, this entity should be taken into consideration in patients with syringomyelia showing acute neurological deterioration.

No MeSH data available.


Related in: MedlinePlus