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��ࡱ�>��	km����j�������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������_�	��0Gpbjbj,E,E	4rN/N/;,��������	�	*****����>>>>�$>�9�&$JJJJ%v�$�?9A9A9A9A9A9A9$�;�B>�e99*%%e9**JJ��9%%%�*J*J?9%?9%%��3|�4J�����V��y������"�W4+9�90�9i46?J$d?$�4�4p?*7����%-t�_���e9e9�$d����9��������������������������������������������������������������������?����������	 �:	POST TRAUMATIC TRANSCALVARIAL BLEPHAROENCEPHALOCELE: A RARE ENTITY
Muhammad Sohail Umerani (MCPS, FCPS),1 Saqib Kamran Bakhshi (MBBS),2 Asad Abbas (MBBS) 3 and Salman Sharif (FRCS)1
1. Department of Neurosurgery, Liaquat National Hospital, Karachi
2. Student, Dow University of Health Sciences, Karachi
3. Department of Surgery, Ziauddin University Hospital, Karachi

Dr. Muhammad Sohail Umerani
Clinic No. 1, Liaquat National Hospital and Medical College, Karachi - 74800
+92 3441 2464
drumerani@yahoo.com

Dr. Saqib Kamran Bakhshi
Clinic No. 1, Liaquat National Hospital and Medical College, Karachi - 74800
+92 3441 2464
saqib_dmc@hotmail.com

Dr. Asad Abbas (Corresponding Author)
J-8, 3rd floor, Yaqoob Terrace, Main Gurumandir Round about, Karachi - 74800
Tel: +92 333 318 3523
Fax: +92 213 586 2940
asad.abbas@live.com

Dr. Salman Sharif
Clinic No. 1, Liaquat National Hospital and Medical College, Karachi - 74800
+92 3441 2464
sharifsalman73@gmail.com
ABSTRACT
Traumatic brain injury is an important cause of morbidity and mortality among pediatric age group, with a male preponderance. Two percent of such conditions are complicated with CSF fistulous leak resulting in meningitis and its sequel. Posttraumatic blepharocele and blepharoencephalocele are unusually rare complications after head trauma secondary to fracture of anterior cranial fossa and fistulous communication between the orbit and subarachnoid space due to ruptured dura. We report such a case in a 5 year old boy after having a fall from roof. To the best of our knowledge this is the second reported case of post traumatic isolated blepharoencephalocele.

Key words: blepharocele, blepharoencephalocele, head injury, post-traumatic






INTRODUCTION
Cerebrospinal fluid (CSF) and brain herniation into eyelid, also known as blepharoencephalocele resulting from head trauma is an exceptionally rare condition. It occurs most commonly in children. We report such a unique case of post-traumatic isolated craniopalpebral fistula. It is the second case of its kind after extensive literature review to the best of our knowledge.
CASE REPORT
A 5 year old boy presented to our out-patient department with complaint of left eye lid swelling after sustaining fall from height of about 10 feet. This was associated with headache without any other symptom.  He was vitally stable with intact higher mental function and no focal or global neurological deficit. There was marked left peri-orbital swelling which was minimally tender, non-pulsatile, non-reducible with normal overlying skin temperature (fig 1). Ocular movements including papillary light reflex and vision in that eye were normal. There was also minimal sub-galeal swelling over left frontal region around hair line about 3-4 cm from mid line.
CT scan brain was advised which showed multiple fractures of left frontal bone extending inferiorly into orbital plate, superior and medial orbital walls with lobulated hypo-dense cystic swelling in left peri-orbital region extending superiorly up to the bony gap. This measured about 4.3 X 1.5 cm2 in peri-orbital region.  
MRI brain was also performed which revealed CSF with brain herniation in to the left upper eyelid through the fracture line with no involvement of the orbit (fig 2). A diagnosis of isolated blepharoencephalocele was thus established. 
He underwent craniotomy and repair of the fistulous communication. Per operatively fractured frontal bone were elevated, dural tear and its edges identified, necrotic brain matter was removed (fig 3). Duraplasty was performed and fibrin glue was used as a sealant (fig 4).
Post operatively the lid swelling gradually started resolving and he was discharged the 2nd day to be followed up in clinic (fig 5). His scalp stitches were removed on 10th post-operative day.
DISCUSSION
Traumatic brain injury is an important cause of disability and fatality in children, frequently resulting from falls, road traffic accidents and play related injuries [1]. Its incidence is twice more common in boys than in girls [2].
CSF leakage is an imperative complication of head trauma, taking place in 2% of all head trauma and in 12-30% of all skull base fractures. Fractures involving the anterior cranial fossa are more commonly associated with it than fractures of the temporal bone [3]. CSF rhinorrhoea and otorrhea are the most common forms of CSF egress after head injury with CSF infrequently migrating into the orbit and is confused with lacrimation [4, 5, 6].
In some cases CSF is retained in the orbit and it is known as orbital encephalocele or orbitocele [7, 8, 9].
Subgaleal post-traumatic herniation of CSF into the eyelids is termed as blepharocele. When there is concomitant herniation of brain substance into the eye lid without involvement of the orbit, it is called blepharoencephalocele. Both of these conditions are exceptionally rare complications of head injury with latter being reported only once previously [4, 10, 11, 12, 13, 14] The cause is a communication between the orbit and subarachnoid space due to fracture of the orbit along with rupture of the dura. 13, 15, 16 Intraorbital blood clot, mucocele, foreign body and orbital abscess are the differentials for such lesions and need to be carefully excluded [15].
While exophthalmos, gaze paresis, oculorroea, or vision disturbances are main features of orbitocele, blepharoencephalocele usually result in bulging of the lid as was seen in our case [10]. CT scan along with water-soluble contrast CT cisternography is a useful investigation showing skull fractures with CSF leaks [15]. However, MRI with gadolinium contrast is the investigation of choice for diagnosing this condition demonstrating brain matter communicating with the eyelid as well as CSF fistulas [15,17,18].
Early clinical and radiological diagnosis of the condition is necessary for prompt management. While it is tempting to incise and decompress the blepharocele/ blepharoencephalocele, this can be hazardous leading to further complications like CSF fistula, bleeding or blephritis. Aspiration or incision of periorbital swelling is therefore contraindicated after head injury. Surgical exploration is considered to be the treatment of choice. This includes craniotomy for excision of non-viable brain tissue, identifying the site of fracture and defining the torn dural margins, and followed by duraplasty along with fistula repair using fibrin sealant. The swelling gradually subsides with good cosmetic outcome [15].
Literature has shown cases described as orbital encephalocele but to the best of our knowledge there is only one reported case of blepharoencephalocele in the past. Such a condition carries a good prognosis if sensibly managed. The surgical treatment not only addresses the fistula but also carries a better cosmetic result.
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Fig 1: Pre-operative inferior, lateral and anterior views of left blephroencephalocele with protruding conjunctiva. There is also evidence of trauma to left forehead.
Fig 2: MRI T2W brain plain showing evidence of a leptomeningeal mass lesion in left peri-orbital region. There is evidence of herniation of brain parenchyma into the left eye-lid.
Fig 3: Peroperative photograph after defining the dural defect and fracture line in the left fronto-orbital region
Fig 4: Peroperative photograph showing application of sealant (Berriplast) over the bone and dural defect
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