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CASE REPORT
Ahead of print publication
 

A rare case of periorbital edema and conjunctival prolapse due to traumatic intraorbital CSF leakage


 Jaiprakash Hospital, Rourkela, Odisha, India

Date of Submission24-Dec-2020
Date of Decision11-Feb-2021
Date of Acceptance11-Feb-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Satya Bhusan Senapati,
Jaiprakash Hospital, Odisha.
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jpn.JPN_328_20

 

   Abstract 

Posttraumatic herniation of brain and CSF into the eyelid is a rare entity with few reported cases in the literature. A 5-year-old girl presented to us with severe periorbital edema and conjunctiva prolapse after 7 days of head trauma. Clinical suspicion of CSF leakage accumulating in lid and periorbita was suspected. CT and MRI scan brain showed multiple comminuted frontal bones, orbital roof, and orbital rim fractures with contused brain tissue herniation through the dural defect. Craniotomy with dural repair was done after which patient improved and within 2 weeks both periorbital edema and conjunctiva prolapse subsided.


Keywords: Blepharocele, conjunctiva prolapsed, skullbase fracture



How to cite this URL:
Panigrahi AK, Senapati SB, Panda D, Panigrahi S. A rare case of periorbital edema and conjunctival prolapse due to traumatic intraorbital CSF leakage. J Pediatr Neurosci [Epub ahead of print] [cited 2022 Dec 6]. Available from: https://www.pediatricneurosciences.com/preprintarticle.asp?id=335204





   Case Report Top


A 5-year-old girl presented to us with severe right periorbital edema and conjunctiva prolapse after 7 days of head trauma. She had history of fall from a height of about 10 feet 7 days back. There was no history of loss of consciousness, CSF rhinorrhea, or focal neurological deficit following fall. After fall there was right periorbital swelling, for which she was admitted to another hospital where she received conservative management with antibiotics and analgesics. Gradually periorbital edema increased leading to conjuctival prolapse. On examination she was conscious and oriented, with no external wounds on the scalp. There was tender, fluctuant, and transilluminant swelling of the right upper eyelid accompanied by chemosis and prolapse of conjunctiva [Figure 1]A. Neuro-ophthalmological examination of the right eye was not possible because of the severe swelling. Visual acuity and external ocular movements were normal on the left side. NCCT of brain showed multiple comminuted right frontal bones, orbital roof, and orbital rim fractures with small underlying brain contusion [Figure 1]B. MRI of brain and orbit showed fracture roof of the orbit, with right frontal base contused brain tissue herniating through undrlying dural defect. CSF was seen leaking through the dural defect and accumulating in the periorbita and eye lid [Figure 1]C. There was no injury to the eyeball or retrobulbar hematoma.
Figure 1: (A) Preoperative picture of patient showing periorbital swelling with conjunctiva prolapse. (B) NCCT of brain showing multiple comminuted right frontal bone, orbital roof, and orbital rim fracture. (C) MRI of brain and orbit showing contused brain tissue herniating through underlying dural defect and CSF accumulating in the periorbita and eye lid. (D) Postoperative day 15 picture showing subsided lid edema and totally reversed prolapsed conjunctiva

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Bicoronal flap with bifrontal craniotomy was made. Through the fractured orbital roof with underlying basal dural and periorbital defect, contused brain matter was found herniating into the upper eyelid. Contused brain was suctioned out, basal dural defect repair done extradurally with pericranial flap. Fibrin glue was used to seal the defect. Bone defects were also repaired with miniplate and screws.

Postoperative recovery was immediate with significant reduction in periorbital swelling noticed within 1 day. On postoperative day (POD) 2, increase in periorbital edema was observed. Tight compression bandage was advised along with lubricating drops. From POD 5, gradual reduction in periorbital edema and conjunctival prolapse was noticed, and by POD 15, significant improvement was noticed [Figure 1D].

Prolapsed conjunctiva was totally reversed and lid edema had subsided. Slit lamp examination revealed normal anterior segment and visual acuity was 6/6 in both eyes. Fundus examination result was within normal limits. Ocular movements were full in all directions of gaze in both eyes.


   Discussion Top


The most common forms of posttraumatic CSF leaks are CSF rhinorrhea or CSF otorrhea. Rarely, orbital fracture with dural injury is associated with leakage of CSF into the orbit. This entity variously termed as blepharocele, blepharoencephalocele, orbital encephalocele.[1],[2],[3] Most traumatic CSF leaks resolve spontaneously, the majority within the first 24–48 h, as a result of blood products and/or inflammatory adhesions at the site of the dural breach and associated skull fracture. In our case, the atypical presentation may be due to the large orbital roof fracture allowing CSF to percolate to the orbital tissues. Since the patient presented to us after 7 days there was a lot of CSF accumulated in the periorbital tissues, which forced the conjunctival tissue to prolapse. Hence a surgical repair was planned. Often this entity is mistaken for posttraumatic ecchymosis. In our case, although the periorbital edema was tense, the overlying skin color was normal. This was a sign which prompted us to think in the direction of CSF leakage accumulating in the lid. Increase in periorbital edema on POD 2 when compared with POD 1 may be due to collection of postoperative blood degraded products in the redundant space created by previous CSF accumulation.

Awareness of this entity is essential to diagnose the condition early. It should be suspected when posttraumatic ecchymosis fails to resolve, especially in patients in whom the superior orbital roof fracture is observed. We came across two case reports which were similar to our case, but the severity and managements were different.[4],[5] In one case there was complete resolution with conservative management only, whereas other patient required orbital roof and dural repair. Our case resembled more to the second case. But one deviation is, in our case, the conjunctival prolapse which worsened after the surgery was managed conservatively, whereas in the second case they went on to drain the pooled CSF through transconjunctival approach.


   Conclusion Top


Although CSF leakage resulting in periorbital edema and conjunctival prolapse is very rare, it should be kept as a differential diagnosis. Proper investigations and neurosurgical intervention may be the cornerstone of management. We should resist from doing drainage of accumulated fluid prior to investigations as it may result in spread of secondary infection through the CSF. To the best of our knowledge, this is a rare presentation of CSF leakage presenting as periorbital edema and conjunctival prolapse successfully managed by combined efforts of neurosurgeon and ophthalmologist as a team.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Chandra N, Ojha BK, Chandwani V, Srivastava C, Singh SK, Chandra A. A rare case of posttraumatic eyelid swelling: cerebrospinal fluid blepharocele. J Neurosurg Pediatr 2013;11:242-4.  Back to cited text no. 1
    
2.
Pease M, Marquez Y, Tuchman A, Markarian A, Zada G. Diagnosis and surgical management of traumatic cerebrospinal fluid oculorrhea: case report and systematic review of the literature. J Neurol Surg Rep 2013;74:57-66.  Back to cited text no. 2
    
3.
Morihara H, Zenke K, Shoda D, Fujiwara S, Suehiro S, Hatakeyama T. Intraorbital encephalocele in an adult patient presenting with pulsatile exophthalmos. Case report. Neurol Med Chir (Tokyo) 2010;50:1126-8.  Back to cited text no. 3
    
4.
Lee J, Kim N, Choung HK, Khwarg SI. Delayed superior forniceal CSF accumulation and conjunctival prolapse after superior orbital wall fracture. Graefes Arch Clin Exp Ophthalmol 2013;251:999-1000.  Back to cited text no. 4
    
5.
Borumandi F. Traumatic orbital CSF leak. BMJ Case Rep 2013. doi:10.1136/bcr-2013–202216  Back to cited text no. 5
    


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