1Department of Neurosurgery, Kyushu Rosai Hospital, Moji Medical Center, Japan
2Department of Neurosurgery, University of Occupational and Environmental Health, Japan
Submission: December 01, 2018; Published: February 07, 2019
*Corresponding author: Takeru Umemura, Department of Neurosurgery, Kyushu Rosai Hospital, Moji Medical Center, 3-1 Higashiminatomachi, Moji-ku, Kitakyushu City, Fukuoka 801-8502, Japan
How to cite this article: Takeru U, Shigeru N, Junkoh Y. Surgical Implication of a Cavernous Hemangioma of the Optic Chiasm ShowingRecurrent Hemorrhage After Long-Term Clinical Stability: A Case Report. Open Access J Neurol Neurosurg. 2019; 9(4): 555769. DOI: 10.19080/OAJNN.2019.09.555769.
Cavernous hemangiomas of the optic chiasm are rare, and hemorrhage from cavernous hemangiomas causes acute visual loss. Although some cases have been reported, the appropriate treatment has not been determined yet because of the scarcity of these reports. Here, we describe a patient with recurrent hemorrhage from a cavernous hemangioma of the optic chiasm after a long clinically stable period, and we discuss the surgical implication of this lesion. A 45-year-old man sustained right visual loss due to hemorrhage from a cavernous hemangioma of the optic chiasm 20 years ago and was conservatively observed. He suffered from acute visual loss in the left eye following sudden onset of headache, and he was hospitalized in our hospital. The patient complained of severe visual disturbance in the left eye in addition to long-term right visual loss. Magnetic Resonance Imaging showed recurrent hemorrhage from the lesion, and the whole chiasm was affected. We performed total excision of the lesion, and his vision improved, enabling him to return to work. This is the first report of recurrent hemorrhage from a cavernous hemangioma of the optic chiasm. We recommend surgical resection, even in clinically stable patients, to prevent recurrence.
Cavernous hemangiomas, also known as cavernous malformations or cavernomas, are vascular malformations characterized by the presence of sinusoid-like capillary vessels [1,2]. They can develop in several locations in the central nervous system, such as in the cerebrum, cerebellum, brainstem, and spinal cord) . However, cases of cavernous hemangiomas involving the cranial nerves are rarely reported. In particular, cavernous hemangiomas involving the optic pathway are extremely rare [4,5]. Therefore, treatment is controversial. Total excision is recommended for most symptomatic patients [6-8]. On the other hand, symptomatic patients may experience improvement after shrinkage of the cavernous hemangioma even without any surgical treatment, and only conservative follow-up is recommended in these cases . Moreover, the surgical indication is obscure in patients with a long-term stable condition after the first episode of hemorrhage, as postoperative visual deficits are associated with decreased activities of daily living.
Here, we describe a patient with recurrent hemorrhage from a cavernous hemangioma of the optic chiasm whose symptoms had remained stable since the initial hemorrhage occurred 20 years ago, and we discuss the surgical indication of the lesion.
A 45-year-old man with acute visual loss in the left eye was admitted. He had a history of hemorrhage from a cavernous hemangioma of the optic chiasm. Since then, he lost central vision loss in the right eye 20 years ago, although left visual acuity was kept intact. Upon admission, he complained of a headache and left visual disturbance in addition to long-term right visual loss. The neuro-ophthalmological evaluation revealed severe left visual disturbance with the preservation of normal pupillary reaction to light in both eyes. The serological examination showed no pituitary dysfunction. Visual disturbance results were as follows: right 0.02 (nc) and left 0.4 (nc). Figure 1 shows the visual field. Visual loss in his right eye was due to previous hemorrhage from the cavernous hemangioma of the optic chiasm, whereas visual loss in his left eye was due to recurrence of the hemorrhage. A computed tomogram showed the hemorrhage in the optic chiasm (Figure 2A). Magnetic Resonance Imaging (MRI) was performed, and an axial Constructive Interference in Steady State (CISS) image showed that the lesion was a
10-mm mass with partial hyperintensity (Figure 2B). On the
coronal MRI CISS image, the patient had multilocular lesions
of different intensities that were found on the right side of the
optic chiasm (Figure 2C). Cerebral angiography did not reveal
any vascular malformations. We considered that the left eye
worsened because the hemorrhage extended over the midline.
The preoperative diagnosis was hemorrhage from the cavernous
hemangioma of the optic chiasm, and we decided to perform
surgical decompression of the optic chiasm.
Surgery was performed via a right pterional approach.
The Sylvian fissure was widely opened, and the optic chiasm
was exposed (Figure 3A). The lesion was brown, and bipolar
coagulation was performed. The cavernous hemangioma shrunk
and was dissected from the optic chiasm (Figure 3B), which was
surrounded by gliosis at the interface. The lesion was totally
removed. The histopathological analysis of the resected lesion
showed vascular spaces containing blood elements, lined by a
single layer of endothelial cells, and the lesion was diagnosed as
a cavernous hemangioma.
The postoperative ophthalmological examination revealed
improvement of visual disturbance in the left eye up to 0.9 (nc)
without changes in the visual field and vision in the right eye.
A cavernous hemangioma of the optic chiasm is rare [4,5].
Some cases have been reported, and the authors recommended
total excision of the cavernous hemangioma of the optic chiasm
[6-8], because early diagnosis is associated with good results
and the prevention of recurrent hemorrhage [9,10]. However,
clinical improvement can occur without operation; thus, close
follow-up was recommended by other authors.
In our case, the patient sustained acute visual loss in the
right eye due to hemorrhage from a cavernous hemangioma
in the optic chiasm 20 years ago, and no clinical change
occurred until the present admission. Recurrent hemorrhage
from the cavernous hemangioma of the optic chiasm occurred
again, and visual function worsened because of the expanding
hemorrhage. If hemorrhage from the optic chiasm occurs
again, visual loss will worsen even if the hemorrhage is small.
Therefore, we consider that surgical treatment is the first
choice for cavernous hemangiomas of the optic chiasm. Even in
clinically stable patients, like ours, the potential for hemorrhage
still exists. According to the literature, vascular malformations
of cavernous hemangiomas of the optic chiasm occur more
frequently than hemorrhages in the brain [10,11]. Therefore,
previous studies support our opinion. We also considered
that excising the hemangioma of the optic nerve could be
conducted without causing neurological deficits; additionally, it
may improve visual disturbance because the gliotic interspace
between the hemangioma and normal neural tissue provides a
plane for dissection, and decompression of the optic nerve may
lead to good results . In our case, we confirmed the gliotic
interspace, and it made excision safe and accurate.
According to the literature, conservative treatment is also
recommended because visual loss may improve after shrinking
the cavernous hemangioma . However, this treatment is not
curative because the hemangioma remains intact and can bleed
again. Visual loss may be severe if hemorrhage recurs. on the
basis of MRI findings, we confirmed that the mass included
lesions of different intensities. Based on the appearance of the
lesions and pathological findings , the different intensities
shown on the MRI CISS image suggested different stages of
hemorrhage. This finding supports the diagnosis of cavernous
In conclusion, cavernous hemangiomas of the optic chiasm,
even in clinically stable patients, need to be totally excised to
prevent recurrent hemorrhage. In addition, MRI CISS imaging is
useful for evaluating the hemorrhage.