|
 Abstract
We report a rare case of multiple
intracranial hydatid cysts in a
12-year-old boy. The patient presented
with nausea, vomiting, gradual loss of
consciousness, anisocoria and
hemiparesis. CT scan showed multiple
cystic lesions in the left hemisphere
and significant midline shift. Immediate
surgical intervention was performed.
Surgical removal of the cysts was
successful and the patient did well
after surgery. No recurrence was
observed during a two-year follow-up
period. Investigation of other organs
showed no involvement.
Key words: multiple hydatid cysts.
cerebral hydatid cysts. echinococcosis
Running head: Multiple cerebral hydatid
cysts
Introduction
Cerebral hydatidosis is an uncommon presentation of
echinococcus species infection in human.
The brain is affected in about 0.5 to 3%
of all cases of echinococcosis.(1,2,3,4,5)
Given the ease of modern travel,
echinococcosis is a worldwide problem
but most of the reports are still from
endemic areas. Cerebral echinococcosis
rarely present as a neurosurgical
emergency. We report an exceptional case
of multiple cerebral hydatidosis
presented with coma where emergency
surgery was performed and the patient
did well after the operation.
Case
Report
This
12-year-old boy referred to our center
with coma. He was a shepherd from a
rural area of Kordestan Iran (an endemic
area of echinococcosis). He had a
history of headache and dizziness for
two months with an episode of tonic-clonic
convulsion about one month prior to
admission. Seven days before admission
he developed nausea, vomiting and
restlessness which was followed by
gradual alteration of consciousness.
He had a history of head trauma
and left-sided epidural hematoma four
years ago for which craniotomy was
performed. He had made a complete
recovery after operation with no
morbidity.
Physical examination revealed normal vital signs and no abnormality on
chest and abdominal examinations. The
patients GCS was 8. He had bilateral
papilledema and right hemiparesis.
Pupils were anisocoric. The left pupil
was 2mm wider than the right pupil. Both
of them were reactive to light. Brain CT
scan showed multiple hypodense cystic
lesions in the left hemisphere with
significant midline shift (Fig-1).
|
Figure 1 . Preoperative CT
scan showing multiple cysts
and significant midline
shift . Note evidences of
the previous craniotomy
which had been performed for
epidural hematoma four years
ago . |
Craniotomy was performed immediately.
Frontal cortex corticotomy was
performed. There was an amorphous
gelatinous material around the cysts.
Gentle dissection between the cysts and
the neural tissue was performed by
spatula, cottonoid and saline
irrigation. All five cysts were removed
intact (Fig-2). The diameters of the
cysts were 2 to 8.5 cm.
|
Figure 2 . Five hydatid
cysts were removed intact . |
The patient did well after
operation and was discharged after two
weeks with no obvious neurological
deficit. Pathological examination
revealed echinococal cyst. Postoperative
brain CT scan showed a mild subdural
effusion with no evidence of
intraparenchymal cystic lesion (Fig-3).
The patient was referred to the
infectious disease department for
further investigation and no evidence of
any other organ involvement was found.
During the two-year follow-up period,
the patient had no evidence of
recurrence.
|
Figure 3 . Brain CT scan one
year after the
operation showing brain
parenchymal changes and mild
subdural effusion without
any evidence of recurrence. |
Discussion
Human hydatid disease, although rare in developed countries,
is a serious problem in some areas of
the world, especially in agricultural
and sheep-raising communities. Human
hydatidosis is an old disease.
Hippocrates, Galen and Avicenna were all
familiar with the condition. Rudolphi
coined the term echinococcus in 1808. In
1890 Graham and Clubb published the
first successful removal of an undoubted
hydatid cyst of the brain.(2)
Hydatid
cyst is an infectious disease in human
caused by the larval stage of
echinococcus granulosus or echinococcus
multilocularis.(2,4,6) Hydatid disease
caused by echinococcus granulosus is
endemic in Iran and most parts of the
Middle East. Liver and lung are the most
common sites of involvement. Cerebral
hydatid cysts are rare. Primary cysts
are more common and usually solitary but
multiple primary cerebral cysts are also
reported in the
literature.(7,8,9,10,11,12,13,14,15)
Secondary cerebral cysts are often
caused by spontaneous, traumatic or
surgical rupture of a primary cerebral
cyst or less commonly due to
embolization of cardiac cysts ruptured
into the left ventricle.(2,3,4,16,17,18)
Both primary and secondary embolic cysts
are commonly found in the cerebral
hemispheres, in the area supplied by the
middle cerebral artery.(3,5)
Hydatid cyst of the brain
is more common in the first two decades
of life and it is more commonly seen in
dwellers of rural countryside in endemic
areas.(2) Symptoms and signs of the
hydatid cyst of the brain are similar to
those of other space occupying lesions;
they are either of general nature due to
raised intracranial pressure or focal
signs due to interference with the
physiological functions of the parts
subjected to local pressure. Hydatid
cyst is a slow growing lesion which does
not invade cerebral tissue but produces
symptoms by exerting pressure due to
increase in its size. Headache, seizure,
enlarged head, visual disturbances and
hemiparesis are common manifestations.
Sensory disturbances, tremor, cerebellar
signs, cranial nerve paralysis and
speech disturbances are less common
presentations of this disease.
Herniation is also a rare presentation
of this disease(2).
Our case was a
child with multiple cerebral hydatid
cyst presenting with cerebral herniation.
Ameli et al described a similar case, a
25-year-old woman with a temporal lobe
hydatid cyst that developed signs of
herniation after a generalized seizure.
Emergency surgery was performed and the
patient made rapid recovery after
operation.(2) The mechanism of
herniation in these patients has not
been described previously. Presence of
the hydatid cysts may cause alterations
in vasoregulatory systems in neural and
glial tissues surrounding the cysts and
lead to edema. Another mechanism may be
acute increase in the size of the cysts
that leads to cerebral herniation.
Head trauma can alter
the pathologic and clinical feature of
the disease during the asymptomatic
stage of the brain hydatidosis but there
is no evidence that trauma could be a
predisposing factor for involvement of
brain with hydatid cyst. This patient
has a history of head trauma and
craniotomy for epidural hematoma four
years prior to involvement of the same
side of his brain with hydatid cysts.
Importance of this event and its
relation with occurrence of cerebral
hydatidosis is not clear for us. Hydatid
cyst should be considered in patients
with cerebral space occupying lesions in
endemic areas. Cerebral herniation may
occur in patients with cerebral hydatid
cysts although it is rare. Emergency
surgical intervention with emphasis on
intact removal of the cysts is
lifesaving in these patients.
References
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