Acta Neurochir (Wien) (2006) 148: 571–578

DOI 10.1007/s00701-006-0751-3

Short Illustrated Review

Cavernous haemangiomas of the anterior visual pathways.
Short review on occasion of an exceptional case

M. Lehner1, F. A. Fellner2;3, and G. Wurm1

1
Department of Neurosurgery, Landes-Nervenklinik Wagner-Jauregg, Linz, Austria
2
Institute of Radiology, Landes-Nervenklinik Wagner-Jauregg, Linz, Austria
3
Institute of Diagnostic Radiology, Friedrich-Alexander-University Erlangen-N€urnberg, Erlangen-N€urnberg, Germany

Received March 21, 2005; accepted January 5, 2006; published online March 2, 2006
# Springer-Verlag 2006

Summary nerves are a rare entity; in extremely rare cases they

The anterior optic pathways are rarely affected by vascular malforma-
tions. In a meticulous literature review, 42 published cases of patients
with vascular malformations within optic nerves, chiasma and=or optic
tract were found, 30 of them being diagnosed as cavernous haemangio-
mas. All of them suffered from visual disturbances; in 38.1% previous
symptoms had occurred. Surgical treatment resulted in major improve-
ment in most patients.
We include a further patient with a cavernous haemangioma of the
optic chiasma and left optic tract who presented with an acute defect of
the right visual field and severe retro-orbital pain. We succeeded in total
excision of the malformation via a neuronavigationally guided approach.
In the postoperative course, vision of our patient improved immediately
and was found to be completely normal three months after the surgical
intervention. Considering our patient and the published cases in the
literature, we are of the opinion that microsurgical excision is a safe
and efficient treatment for these rare pathologies.
Keywords: Cavernous haemangioma; anterior optic pathways;
cranial nerves; microsurgical excision.
Introduction
Cavernous haemangiomas are angiographically occult
vascular malformations which may occur throughout the
body and neuroaxis, and affect 0.3–0.7% of the popula-
tion [6, 33]. The most common location of cavernous
haemangiomas of the central nervous system is the
supratentorial subcortical white matter. They may also
occur in the cerebellum, the brainstem, the spinal cord,
or the spinal nerves [33]. The percentage of distribution
reflects the volumes of the distinct compartments of the
central nervous system [6]. Thus, cavernomas of cranial
have been reported to affect the anterior optic pathways
[1, 3–7, 9, 10, 12, 14–17, 20, 22–25, 28–31]. Even more
rarely, other vascular malformations like venous angio-
mas [2, 8, 11, 13] and arteriovenous malformations
[11, 17, 18] have been found at this location. Vascular
malformations within the anterior visual pathways can
cause visual symptoms as well as headache, retro-orbital
pain and nausea. Clinical symptoms are mostly due to
intrinsic or extrinsic bleeding.
We undertook a thorough review of the literature on
the topic of cavernous haemangiomas and other vascular
malformations affecting the anterior optic pathways,
where we focused on clinical presentation, imaging
modalities, localisation, histopathology and therapeutic
strategies (Table 1). The review was initiated by a
patient, who presented with acute visual dysfunction
and retro-orbital pain caused by bleeding of a cavernous
haemangioma located in the chiasm and in the left optic
tract.
Literature review
The pubmed search engine of the national library
of medicine and national institutes of health (www.
pubmed.gov) was used to perform an internet medline
literature research based on the search items ‘‘chiasma’’,
‘‘cavernoma’’, ‘‘vascular malformation’’ and ‘‘cranial
nerves’’. No limitations for the timeframe, language
Table 1. Review of literature on vascular malformations of the anterior optic pathways: clinical and surgical features
572

Author Year n Sex Age Symptoms Prev. sympt. Onset Imaging Bleeding Therapy Histology Outcome

Fermaglich [8] 1978 1 m 30 V, P, N N subacute CT, AG Y decompression, biopsy VA improved

Carter [2] 1982 1 f 48 V Y acute CT, AG Y total excision VA improved
Maitland [19] 1982 3 m 26 V, P, N Y acute CT, AG Y decompression, biopsy n.r. improved
f 23 V, P N acute CT, AG Y decompression n.r. recovery
f 63 V, confusion Y chronic CT, AG Y decompression, biopsy n.r. improved
Lavin [17] 1984 3 m 37 V, P Y chronic CT, AG Y 2 decompression AVM improved
m 43 V, P N acute CT, AG Y decompression CAV improved
f 27 V, P N acute CT, AG Y decompression AVM improved
Mohr [23] 1985 1 m 30 V, P, confusion Y acute CT, AG Y decompression, biopsy CAV improved
Hankey [11] 1987 2 f 36 V, P N acute CT, AG Y decompression, biopsy AVM improved
m 26 V N acute CT, AG Y decompression, biopsy VA n.r.
Hufnagl [13] 1988 1 f 39 V Y acute CT, AG Y decompression, biopsy VA improved
Marouka [22] 1988 1 f 24 V N subacute CT, AG Y total excision CAV unchanged
Tien [30] 1989 1 f 32 V Y n.r. CT, AG N biopsy, radiotherapy CAV improved
Hassler [12] 1989 3 f 24 V, P Y acute CT, MRI, AG Y total excision CAV improved
m 16 V N chronic CT, MRI n.r. total excision CAV deterioration
m 35 V, P N acute CT, MRI, AG Y total excision CAV improved
Corboy [5] 1989 1 f 44 V, P Y acute CT, AG Y biopsy CAV unchanged
Regli [25] 1989 1 f 28 V, P N acute CT, MRI Y total excision CAV improved
Castel [3] 1989 1 f 23 V, P N subacute CT, MRI, AG Y total excision CAV improved
Lejeune [18] 1990 1 f 26 V, P N acute CT, MRI Y decompression, biopsy AVM improved
Steinberg [29] 1990 2 m 58 V, P, CN VI-palsy N acute CT, MRI, AG n.r. biopsy CAV improved
f 33 V, P Y progressive MRI, AG Y laser resection CAV improved
Malik [20] 1992 1 f 4 V N progressive CT, MRI Y partial resection CAV unchanged
Ferreira [9] 1992 1 m 8 V, P, N Y acute CT, AG Y total excision CAV unchanged
Hwang [14] 1993 1 m 42 V Y acute CT, MRI, AG Y decompression, biopsy CAV improved
Shibuya [28] 1995 2 f 18 V, P Y chronic CT, MRI N total excision CAV improved
f 60 V, P N subacute CT, MRI, AG Y total excision CAV improved
Warner [31] 1996 1 f 32 V, P N acute MRI Y total excision CAV improved
Arrue [1] 1999 3 n.r. n.r. V, P N acute CT, MRI Y observance n.r. recovery
n.r. n.r. V, P N progressive CT, MRI Y partial resection CAV improved
n.r. n.r. V, P Y acute CT, MRI Y total excision CAV improved
Iwai [15] 1999 1 f 31 V, P N acute MRI, AG Y total excision CAV improved
Christoforidis [4] 2000 1 m 38 V, P N subacute CT, MRI, AG Y n.r. CAV n.r.
Elmaci [7] 2000 1 f 28 V, P N acute CT, AG Y total excision CAV improved
Paladino [24] 2001 1 f 58 V, P N chronic MRI Y total excision CAV improved
Glastonbury [10] 2003 1 f 25 V, P N acute MRI Y total excision CAV n.r.
Kehagias [16] 2003 1 m 27 V, P Y chronic MRI n.r. total excision CAV n.r.
Deshmukh [6] 2003 4 m 34 V n.r. subacute n.r. n.r. total excision CAV improved
f 29 V n.r. acute n.r. n.r. total excision CAV improved
f 28 V Y progressive MRI n.r. total excision CAV improved
f 29 V n.r. acute n.r. n.r. total excision CAV improved
M. Lehner et al.

AG angiography, AVM arteriovenous malformation, CAV cavernous haemangioma, CN cranial nerve, N nausea, n.r. no remarks, P pain, Prev. sympt. Previous symptoms, V visual, VA venous angioma.
Cavernous haemangiomas of the anterior visual pathways
and publication type were imposed on the search pro-
cess. The first relevant article was published in 1978 by
Fermaglich [8]. Full papers of all hits, dealing with
the correct localisation and pathology, were ordered.
Articles were available in the English, French and Italian
languages. The references of the full articles were com-
pared with the positive responses of our search in order
to complete the literature table. We found 28 relevant
case reports and short reports dealing with the topic of
vascular malformations of the anterior visual pathways
[1–20, 22–25, 28–31]. Double publications were ex-
cluded. Most authors reported on a single case [2–5,
7–10, 13–16, 18, 20, 22–25, 30–31], eight reported on
2 to 4 cases [1, 6, 11–12, 17, 19, 28–29]; five publi-
cations enclosed a literature search and review on
‘‘chiasmal apoplexy’’, ‘‘cryptic chiasmal malforma-
tions’’, or ‘‘cavernous haemangiomas of cranial nerves’’
[6, 17, 18, 28, 31].
In total, 42 cases of vascular malformations involv-
ing the anterior optic pathways were found, 30 of them
being histologically diagnosed as cavernous haeman-
giomas (Table 1). Published data were reviewed accord-
ing to age, sex, symptoms, acuteness, lesion localisation,
imaging, therapeutic strategies, histopathology, and
outcome.
Analysis of literature review
The mean age at clinical presentation of the whole
patient series was 32.4 years with an age range of 4 to
63 years. The ratio between males and females was 14 to
25; in three cases there was no comment on sex and age.
The most common clinical symptoms and signs of
the patients reviewed were visual disturbances (100%,
42 patients), headache and=or retro-orbital pain (69%,
29 patients). Sudden onset of complaints (59.5%, 25
patients) was observed more frequently than subacute,
chronic or slow progressive development of symptoms.
In 16 of 42 reviewed patients (38.1%), previous episodes
of typical symptoms had occurred before a correct diag-
nosis was made. The optic chiasma was involved in
38 patients (90.5%), whereas sole location in the optic
nerve or in the optic tract was found in only one
case, respectively. Bleeding was detected in 78.6%
(33 patients) (Table 1).
Surgical treatment was performed in 40 of 42 reported
patients (95.2%), clinical observance without surgical
intervention was chosen in one case [1], no remarks
on therapeutic strategy could be found in the other case
[4]. The surgical techniques used varied from total exci-
573
sion in 22 patients (55%), partial resection in two cases
(5%), decompression with biopsy in 13 patients (32.5%),
simple biopsy in two cases (5%), biopsy and additional
radiotherapy in one case (2.5%).
80% (32 patients) of the surgically treated patients
experienced improvement of symptoms, including one
patient with total recovery after decompressive resec-
tion [19]. In 10% (4 patients), symptoms remained
unchanged. Only one patient experienced deterioration
after total excision of the malformation [12]. There were
no remarks on outcome in three patients (Table 1).
Histological examination of the specimen of patients
reviewed proved the lesions to be cavernous haemangio-
mas in 30 patients (71.5%), venous angiomas in four
patients (9.5%) [2, 8, 10, 12] and arteriovenous malfor-
mations in four patients (9.5%) [10, 16, 17]. In four
cases, no information about histopathology was avail-
able [1, 19] (Table 1).
Illustrative case
A 39-year-old female patient presented with acute
defects of the right visual field and severe retro-orbital
pain. The patient had a long history of headache; on
clinical examination no other neurological deficit could
be found. Computed perimetry showed partial hemia-
nopsia of the right visual field. Preoperative visually
evoked potentials demonstrated loss of usual configura-
tion of the potential for the stimulated left eye with
deformed and delayed triple peak of diminished ampli-
tude. No major abnormality was found for the stimulated
right eye.
The CT-scan showed multiple hyperdense struc-
tures without contrast enhancement in the left frontal
lobe, in the pons and within the optic chiasm. Pre-
and postoperative MR examinations were done using a
1.5 T whole body scanner with a standard head coil
(Magnetom Symphony, Siemens). MR protocol included
T2-weighted turbo spin-echo (TSE), T2 -weighted
gradient-echo (GRE), T1-weighted conventional spin-
echo sequences before and after i.v. application of
gadolinium (Omniscan, Amersham Health), dosage
0.1 mmol=kg body weight, in the transverse orientation
(slice thickness 6 mm, gap 1.2 mm) as well as a contrast
enhanced T1-weighted 3D MP-RAGE data set with an
effective slice thickness of 1.0 mm. MRI demonstrated
the supra- and infratentorial lesions to be cavernous
haemangiomas, suggesting a familial pattern. The lesion
corresponding to the neurological deficit was the one in
the chiasm reaching into the left optic tract which
574 M. Lehner et al.

showed typical features of acute intrinsic haemorrhage
(Fig. 1a). MR-angiography showed no pathological
features.
Microsurgical neuronavigation-guided excision was
performed via left pterional craniotomy. Most of the
cavernoma was situated within the optic chiasm and
the left optic tract (Fig. 2a) and a small part was
reaching up to the surface (exophytic portion). Meticu-
lous dissection was performed in order to achieve total
excision of the malformation and to leave the sur-
rounding haemosiderin-stained tissue intact (Fig. 2b).
Intra-operative visually evoked potentials did not show

Fig. 1. Magnetic resonance imaging of a cavernoma located in the optic chiasm extending into the left optic tract. Preoperative imaging (a–e, g):
(a) T2-weighted turbo spin-echo, (b) T2 -weighted gradient-echo, (c) conventional spin-echo before and (d) after i.v. use of gadolinium (Omniscan)
as well as (e) transverse, and (g) coronal thin slice (1 mm) reconstructions from a gadolinium enhanced 3D MP-RAGE data set. T2-weighted
turbo spin-echo (a) shows the typical ‘‘popcorn’’ aspect of a cavernoma. T2 -weighted GRE reveals typical ‘‘blooming’’ of the lesion due to
calcifications=haemorrhage. There is no enhancement after i.v. gadolinium (c, d). Reconstructions of the 3D data set show the localisation of the
cavernoma within the optic chiasm, extending into the left optic tract. Postoperative imaging (f, h): transverse and coronal reconstructions from the
3D data set reveal postoperative reactive changes after complete removal of the cavernoma
Cavernous haemangiomas of the anterior visual pathways
Fig. 1 (continued)
any changes during the operation. Histological exami-
nation proved the lesion to be a partially throm-
bosed cavernous haemangioma with signs of recurrent
bleeding.
The postoperative course was uneventful, vision im-
proved rapidly. Three months after the surgical interven-
575
tion, the patient had normal vision, and MRI proved total
removal of the cavernous haemangioma (Fig. 1b). We
also offered surgery for the cavernomas in the pons,
which also showed signs of recurrent haemorrhage, how-
ever, the patient has so far refused surgery for these
lesions.
576
Fig. 2. (a) Microsurgical view: a small portion of the cavernoma is reaching up to the surface of the chiasm (exophytic portion); Neuronavigation
with contour guidance shows the major mass of the cavernoma lying within the optic chiasm reaching into the left optic tract. (b) Microsurgical view
after complete resection of the cavernoma
Discussion
Clinical symptoms and signs
The natural history of cerebral cavernomas is still a
matter of debate [26, 27]; bleeding rate is mostly
quoted 0.7% per year [28], rebleeding rate seems to be
much higher and may go up to 4.5% per year [24].
Because of the eloquence of tissue, vascular malforma-
tions of the anterior visual pathways pose a significant
threat to visual function. Sudden onset of headache
combined with acute visual loss caused by acute bleed-
ing in the optic chiasm was first described in this
context by Maitland et al. [19]; they used the term
‘‘chiasmal apoplexy’’ for this kind of clinical presen-
tation. In most patients of our series, bleeding was
observed (78.6%). Thus, bleeding frequency at this loca-
tion seems to be much higher than at others (Table 1).
However, this phenomenon may actually be due to the
fact that even very subtle bleeds are prone to cause
symptoms when the lesion is lying within cranial nerves
[6, 28].
Analysis of the data available in literature showed
that 38.1% of the reviewed patients had been suffer-
ing from clinical symptoms for many years. Our patient
also reported on chronic headache, but she had no
history of visual disturbances. Acute visual dete-
rioration and retro-orbital pain occurred at an age of
M. Lehner et al.
39 years and was due to acute intrinsic haemorrhage
of the lesion.
Imaging
Published data prove MRI to be the investigation of
choice for all locations of cerebral cavernous malforma-
tions [1, 28–30]. MRI is especially sensitive in small
anatomical structures like the cranial nerves [28].
Moreover, MRI provides fundamental data concerning
intrinsic or extrinsic bleeding or growth of vascular mal-
formations. Since signals vary according to the stage of
haemorrhage and according to variations in oedema
around the lesion [24], MRI allows monitoring of a
lesion’s evolution and follow-up studies. Especially gra-
dient echo-sequences (T2 weighted images) are very
sensitive in revealing small haemorrhages and calcium,
and are thus superior to spin-echo and conventional
T2-weighted scan [32].
An interesting detail in our literature review consists
in the fact that in the pre-MRI era conventional angio-
graphy was performed in all reported cases [2, 8, 11, 13,
17, 19, 22, 23, 30]. Since MRI is available, supplemen-
tary angiography has been performed in only 42.9%
(Table 1). In none of these cases could additional infor-
mation be obtained with the help of conventional angio-
graphy. We prefer MR angiography, as it seems to be
Cavernous haemangiomas of the anterior visual pathways
sufficient to exclude associated vascular anomalies and
due to the fact that it is a non-invasive investigation.
Therapeutic strategies and outcome
In 80% (32 patients) of the surgically treated patients,
improvement of symptoms was reported, including one
patient where total recovery was seen after decompres-
sive resection [19]. In 10% (4 patients), symptoms
remained unchanged after the operation. We found no
remarks on outcome in three patients. Only one patient
experienced deterioration after total excision of the mal-
formation [12]. Our patient is the second case in the
literature, where total recovery of vision occurred after
surgery, and the first with total recovery after complete
resection. As demonstrated in our case and in previous
reports, vascular malformations of this region can be
safely resected with preservation or even restoration of
cranial nerve function.
Incomplete resection may occur when vision of the
operative field is restricted. But remnants of the malfor-
mation can lead to re-growth of the remaining lesion and
to recurrent bleeds [6]. Therefore, the attempt at total
resection has to be recommended. To achieve a satisfac-
tory result it has to be stressed that particular attention
has to be paid to careful microsurgical dissection. In this
context, neuronavigation is a useful tool to ensure total
resection of ‘‘invisible’’ parts of cavernous haemangio-
mas when they are lying with in cranial nerves and only
a small portion is reaching up to the surface.
Neuromonitoring of cranial nerves during the resec-
tion of cavernous haemangiomas increases the safety of
the procedure and can reveal neuronal dysfunction at a
very early stage. Additionally, we recommend the usage
of intra-operative visually evoked potentials during sur-
gery in and around the optic pathways, i.e. optic nerve
gliomas, skull base meningiomas, craniopharyngiomas,
pituitary tumours and giant aneurysms.
In contrast to locations with minor eloquence [32],
resection of surrounding haemosiderin-stained tissue is
not advisable in cranial nerve cavernomas.
In conclusion, our patient and the reported cases in
literature show that cavernous haemangiomas of the
anterior visual pathways have typical clinical charac-
teristics, and should therefore lead to prompt imaging
and correct diagnosis. Additionally, the intervention of
choice should be surgical treatment aiming at total
resection. The postoperative course of our patient shows
that total recovery of visual function is possible after
microsurgical neuronavigationally guided resection.
577
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Comment
Dr. Lehner and his colleagues present a unique case of cavernous
hemangioma involving the optic chiasm as well as the left optic tract.
The patient was managed surgically with complete resection of the
cavernous hemangioma.
Authors in their meticulous review of the literature found 42 pre-
viously reported patients with vascular malformations within optic
nerves, chiasm, or optic tracts, 30 of them being cavernous hemangio-
mas. Visual field deficits were present in all of these patients with acute
onset in majority of them.
Presented information clearly points towards surgery as a preferred
treatment strategy. Rareness of this entity will most likely prevent us to
find definite answer, but microsurgical interventions mainly in acute
onset of symptoms seems to be justified.
Stephen E. Griffith and Roman Hlatky
San Antonio, Texas, USA
Correspondence: Michael Lehner, Department of Neurosurgery,
€ . Landesnervenklinik Wagner Jauregg, Wagner-Jauregg-Weg 15,
O. O
A-4021 Linz, Austria. e-mail: michael.lehner@gespag.at