British Journal of Ophthalmology, 1986, 70, 570-574

Pseudoepiphora from cerebrospinal fluid leak:case report

ROBERT M DRYDEN,' AND ALLAN E WULC2

From the 'Department of Ophthalmology, University ofArizona, Tucson, Arizona, and2University ofPennsylvania Scheie Eye Institute, Department of Ophthalmology, Philadelphia,Pennsylvania, USA

SUMMARY A 4-year-old tearing child with obstruction of the nasolacrimal duct was treated withdacryocystorhinostomy three years after naso-orbital injury. However, what appeared to betearing peristed, and meningitis developed. Coronal CT scans demonstrated traumatic encepha-locele of the posterior superior orbital roof. A chronic orbital cerebrospinal fluid (CSF) leak wasdiagnosed. To our knowledge no case of chronic CSF leak has been reported that simulated tearingin an otherwise asymptomatic child. In the tearing patient who has a naso-orbital fracture thepossibility of chronic CSF leak should be considered.

Lacrimal outflow obstruction is a well recognisedcomplication of midfacial fractures involving themaxilla, the lacrimal bones, or the ethmoids.18Common manifestations of obstruction to thelacrimal outflow system include epiphora, dacryo-cystitis, or mucocele.A case of epiphora thought to be secondary to

nasolacnmal duct obstruction following midfacialtrauma is herein described. A 4-year-old child seentwo years after naso-orbital injury had traumaticlacrimal duct obstruction and underwent uncompli-cated dacryocystorhinostomy (DCR).The aetiology of tearing in this child was chronic

post-traumatic cerebrospinal fluid (CSF) leakagethat occurred simultaneously with lacrimal ductobstruction. Despite a successful, DCR, chroniccopious CSF leakage simulating tearing continued.To our knowledge this is the first reported case ofpseudoepiphora caused by chronic CSF leakage in anotherwise asymptomatic child.

After midfacial fracture chronic CSF leak shouldbe considered in the differential diagnosis of tearing,and the appropriate laboratory and radiographictests should be performed.

Case report

A 4-year-old boy was seen with epiphora of the righteye of two years' duration. A motor-car accident twoCorrespondence to Robert M Dryden, MD, 601 N Wilmot Road,Suite 58, Tucson, Arizona 8571 1, USA.

years previously resulted in hospital admission,where lethargy progressing to coma and blood in themiddle ear led to the diagnosis of basilar skullfracture. Computerised axial tomography (CT) wasdone three times while he was in hospital and thescans were reported as normal.

Immediately following hospital discharge persis-tent right-sided tearing was noted. There was noirrigation or pain, and tearing was not related toeating or salivation. Ocular examination showedvisual acuities of 20/30 in both eyes. Right inferioroblique overaction was noted with a V-patternexotropia. Palpation of the orbit revealed no anteriordefects and a normally positioned, non-pulsatileglobe with no rim defects.

Lacrimal examination with a Jones primary dyetest showed no passage of dye after 40 minutes.Traumatic lacrimal drainage obstruction was in-ferred.An examination under anaesthesia revealed an

obstructed nasolacrimal duct. No fluid could beirrigated through either the upper or lower canalicu-lus into the nose. Probing revealed a tight internalcommon punctum and an obstructed nasolacrimalduct.A dacryocystorhinostomy with canalicular intuba-

tion with silicone tubing was performed. The bonewas thickened, presumably from past naso-orbitalfracture. The patient received 500 mg of intravenouscephazolin (Kefzol) and oral cephalosporins werecontinued for seven days.

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On the fifth postoperative day mild erythema ofthe scar site was noted. Tearing had not ceased.One week after operation the patient developed

fever with headache and vomiting. He was admittedto a community hospital with a presumptive diagnosisof periorbital cellulitis and begun on intravenouscephazolin. Two days later he complained of neckpain. A lumbar puncture was done which showedcloudy CSF, raising the possibility of bacterial menin-gitis. An additional antibiotic was instituted. An axialCT scan was performed which showed air and softtissue swelling of the right orbit consistent with theoperative appearances as well as an opacity of theright ethmoids consistent with the postoperativehaemorrhage he had had.

In the ensuing two weeks the patient developedrecurrent spiking fevers accompanied by meningealsigns. Intravenous antibiotics were altered to meth-icillin and chloramphenicol, and he remained afebrileuntil three weeks after operation, at which timeanother temperature spike to 101 degrees was noted.The patient was transferred to Tucson MedicalCenter for further evaluation.On admission, the temperature was 38 3°C and the

vital signs were otherwise normal. No periorbitalcellulitis was noted. The dacryocystorhinostomy scarwas healing well. The silicone stent was in place.Tearing was persistent and the results of the eyeexamination were otherwise normal. The medicalexamination, including a neurological examination,gave results within normal limits. All laboratory testsincluding examination of the CSF gave normalresults.On the second day in hospital an ophthalmic plastic

surgery opinion was sought. The child continued to

Fig. 1 Coronal CTdemonstrating DCR ostium andthickening ofnasal mucosa on the right (arrow).

'tear' profusely in the right eye. A Jones primary dyetest showed dye in the oropharynx at 2 minutes. Itwas concluded that the dacryocystorhinostomy sur-gery had been successful and that causes of pseudo-epiphora should be sought to explain the 'tearing'.Our examination included a Dextrostix test of the

tear fluid. The sugar content of the tear fluid wasobserved. The Dextrostix test of the right eye showeda glucose level of 100 to 180 mg/dl (5-6-10.0 mmol/l).The glucose level from the left eye was less than 100mg/dl (5.6 mmol/l). A specimen of the patient's tearswas collected and sent for laboratory analysis. Adiscrepancy of 2 to 1 in the glucose concentrationexisted in the right and left eyes. The data stronglysuggested CSF leakage into the orbit. A CT scan wasperformed (Figs. 1 and 2). A gas lucency in the regionof the right orbit with thickening of the right medialrectus and opacification of the right ethmoid wasobserved. Anteriorly the DCR ostium was visiblealong with thickening of the nasal mucosa (Fig. 1). Abony defect was noted in the posterior orbit thatdid not communicate with the DCR ostium (Fig.2).On the tenth day in hospital a frontal craniotomy

was performed. An encephalocele wasfound into afracture in the posterior medial orbital roof. It wasretracted from the fracture site, and a Silastic implantwas placed over the defect. The dural tear was notedand repaired with a free pericranial graft and closedwith a single running suture. Postoperatively thepatient did well and was discharged six days later.Tearing ceased immediately after surgery. Measuredglucose levels in both eyes were the same and lessthan 100 mg/dl (5.6 mmol/l).

'demonstrating defect in orbital roofonthe rignt (arrows).

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Discussion

CSF leak has been reported to occur in 6% to 35% ofpatients with severe non-penetrating intracranialinjury.'59 It occurs in approximately 20% of patientswith midfacial fracture or fractures through theparanasal sinuses.:CSF leaks are rare in children below the age of 2

because of the flexibility of the cranial base and therelative immaturity of the paranasal sinuses. How-ever, in children older than 2, CSF leakage is by nomeans uncommon."'Traumatic CSF leaks usually result from tears in

the dura at the skull base that allow CSF to escapefrom the subarachnoid space into the nasopharynx,the paranasal sinuses, or subcutaneously. The floorof the anterior cranial fossa, particularly the cribri-form plate area, is thin, and the dura at this site firmlyinvests the olfactory fissure where the olfactory nervepenetrates the skull. Midfacial trauma may result inpenetration of the anterior cranial fossa by ethmoidalroof or wall fragments and dural tear. CSF escapesimmediately. If the edges of the dura are in apposi-tion and do not gape, healing is relatively rapid andCSF leak resolves in three to 10 days. Sinus mucosamay bridge a gaping dural defect and cause leakage tocease. However, if bony spicules project through thetear, a portion of the brain herniates through thedefect. If a large dural defect is present, healing maybe incomplete. High CSF flow due to coughing orstraining may also result in late onset of CSF leakagedue to rupture of a partially healed tear. Dural tearsmay be enlarged by brain and high amplitude CSFpulsation with resultant erosion of bone.9 "The diagnosis, aetiology, and management of

fractures of the orbital roof have been well des-cribed by McClachan et al. 2 Rhinorrhea is themost common presenting symptom of CSF leakageand presents in 25% of patients with midfacialfracture.' Hypo-osmia or anosmia may occur fromdamage to olfactory nerve fibres in 5% of patients.9Smell may be intact in patients who have anteriorlyplaced frontal sinus fractures or sphenoidal sinusfractures. If fracture of the petrous portion of thetemporal bone has occurred, CSF from the middle orposterior cranial fossa may escape into the mastoidsand produce middle ear fluid mimicking serous otitismedia. Rarely CSF tracks subcutaneously, producinga subepicranial hydroma.'3 Headache occurs in 15%of patients with CSF leakage and is therefore thoughtto be an inconstant symptom.9 Meningitis occurs inbetween 3% and 50% of cases ofCSF leakage and theincidence is higher when the leakage is chronic."̀116The observant patient may describe a gush of salty

fluid, or choke on arising in the morning. In therecumbent position leaking CSF collects dependently

in the sphenoid sinus. On the patient's standing, aninflux of CSF collected overnight rushes into theoropharynx causing the so-called reservoir sign.'7

Other signs of CSF leakage include frequentawakening from sleep with coughing fits and soakingof the bed sheets with clear nasal discharge eachnight. Rhinitis has also been described from chronicCSF leakage.9"CSF leakage following midfacial fracture in an

otherwise asymptomatic patient may be confusedwith traumatic epiphora. In a series of 300 patientswith midfacial fracture where 35% had CSF leakage,5 3% had concomitant epiphora or dacryocystitis.5 Ina series of 100 patients reported by Campbell withmidfacial fracture 12% had complaints of tearing.'8Naso-orbital trauma may injure the medical pal-pebral tendon, lacerate or compress the nasolacrimalsac or duct, or cause cicatricial ectropion of the lowerlid or punctal malposition.478 While CSF leakageusually resolves, dacryocystitis and epiphora com-monly persist until surgically treated.Few cases of chronic CSF leakage into the orbit

have been reported in the neurosurgical or oph-thalmic literature. In a review of giant epidermoidand dermoid tumors of the orbit Carey described twocases of chronic CSF leakage into the orbit thought tobe from direct communication between the subarach-noid space and the inner portion of the tumour. 9 Inhis series other symptoms including pulsating prop-tosis, and extraocular movement limitation were alsoobserved. 9A case of traumatic CSF fistula simulating epiphora

occurred in a child 8 months of age and was reportedin the neurosurgical literature.2" Epiphora occurredimmediately following head trauma. The patientpresented with coma, hemiparesis, and facial weak-ness and developed a pulsating exophthalmos accom-panied by bruit and chronic epiphora. Chemosis andepiphora resulted from a leak through the ethmoidalsinuses directly to the conjunctiva. The child under-went surgical repair of the fistula soon afterwards.A case of CSF fistula in a 22-year-old victim of a

motor vehicle accident with a cranial nerve palsy hasbeen reported. This patient had a pulsatile rightupper lid and chemosis from direct subarachnoidcommunication with the upper lid. No tearing wasobserved at any time.2'The present case of CSF leakage differs from the

above-mentioned cases and has many interesting andhitherto undescribed features. It occurred chroni-cally and continuously in an awake and alert child fora period of three years following injury. Proptosis,chemosis, or eyelid swelling were not noted at anytime, and neurological deficit did not develop. Nopulsations were observed. Finally, tearing was notsuspected to be secondary to orbital CSF leakage

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until after successful lacrimal drainage surgery hadbeen performed.CSF leakage occurred as a result of dacryocystor-

hinostomy surgery in two cases reported by Neuhausand Baylis and may occur if bone removal extends tothe level of the cribriform plate.22 The same authorsnoted an average of 5 mm between the nasal ostiumand the floor of the anterior cranial fossa.22 While it ispossible that in the reported cases DCR surgerycould have created a CSF leak, it is unlikely. Tearingwas chronic prior to surgery and persisted aftersurgery, and it promptly resolved following neuro-

surgical repair of the posterior orbital encephalocele.Other unlikely hypotheses that might explain thefindings in the presented case include the possibilitythat CSF leaked from the canaliculi in retrogradefashion from the abnormality of the cranial vault,simulating tearing. The CT evidence for this theory islacking, as the encephalocele was well posterior tothe lacrimal drainage system. While remanipulationof a traumatically altered orbit and cranial baseundoubtedly introduced meningeal infection, thepathway of CSF leakage from posterior orbit to thetear film is a matter of speculation.

Reagent strips such as Dextrostix and Labstixcommonly used for urine glucose determination,while not helpful in distinguishing CSF from nasaldischarge,'223-26 may be helpful in diagnosing CSFleakage into the orbit. CSF glucose is approximately70% of that of serum glucose. Normal levels ofglucose in tears are insignificant.26 27 For the purposesof the clinical examination in the reported case, tearconcentration in 20 normal individuals without dia-betes mellitus was measured with a Labstix reagentstrip by placing it directly on the tear meniscus andwaiting until wetting had occurred. After 30 secondsthe glucose level was measured by matching thecolour of the paper with the colour of the glucose asdetermined on the table found on the bottle. In allpatients the glucose level was found to be within 0and 100 mg/dl (0 and 5 6 mmol/l). In the casedescribed here the glucose level on the uninvolvedside was from 0 to 100 mg/dl, corresponding to thenormal value. On the involved side the tear glucoselevel was between 100 and 180 mg/dl (5.6 and 10-0mmol/l). The higher glucose level on the involvedside in this patient was confirmed by laboratoryanalysis. A large sample needs to be collected forglucose concentration to be analysed in a laboratory.The reagent strips are inexpensive, immediate, andrequire substantially less tear volume and were

effective in diagnosing the leakage.The site of CSF leakage may be localised radio-

graphically with or without the aid of contrastmaterial. Skull films may show an air fluid level in thesinuses.9 Materials can be instilled intrathecally to

demonstrate leakage of CSF. Methylene blue andfluorescein were at one time injected but have beenabandoned owing to numerous complications."2"29Radioiodide labelled serum albumin and technetiumare used to directly visualise CSF leak with cistern-ography and are diagnostic in 86% of cases."'Metrizamide encephalocistemography with highresolution CT is mandatory in evaluation of thepatient with a suspected chronic orbital CSF leak. Inour patient CT scans with axial views were obtainedat the time of the original trauma and repeatedlythereafter to evaluate brain, orbits, and skull baseand were all within normal limits. One week follow-ing DCR axial CTs again did not show any abnor-mality of the skull base or orbital roof. However,coronal CTs with 1-5 mm cuts clearly show a pos-terior bony defect and suggested meningoencephal-ocele and chronic CSF leak. The different perspectiveoffered by directly formed coronal views may be anasset in considering the possibility of chronic CSFleakage into the orbit.When a CSF leak is noted in the early period post

midfacial injury, opinions are varied as to whetherdirect and immediate reduction of facial fracture orrepair of dural tear is indicated. Most authorsrecommend bed rest with the head of the bed raised.Prophylactic antibiotics are administered intra-venously. Bed rest, stool softeners, and medicationto prevent coughing or straining are recommended.A lumbar drain, placed in the lumbar subarachnoidspace, may be useful in lessening CSF flow throughthe dural rent, thus allowing repair to take place.Likewise, acetazolamide may be administered todecrease the production of CSF.The majority of CSF leaks heal without the need

for surgery.9" When the neurological condition hasstabilised, fractures may be reduced with therealisation that additional trauma to the dura mayresult.The treatment of chronic persistent CSF leak is

neurosurgical."I 1617 If meningitis has supervened, anadequate period of time should elapse for the patientto be on appropriate antibiotics. Chronic CSF leaksusually do not subside spontaneously. The area of thedural rent must be localised and repaired. In thereported case a frontal craniotomy with exposure ofthe fracture site, Silastic roof implant, and epicranialduroplasty were performed. The techniques of thisneurosurgical procedure go beyond the scope of thispaper.

Patients who tear after naso-orbital trauma mustbe suspected of having a CSF leak even if signs ofnasolacrimal duct obstruction are present. It may beuseful to ask patients specifically about rhinorrhoeaor anosmia, as well as to examine for increasedtearing during the Valsalva manoeuvre. CT scans

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should be reviewed if they have been performed andcoronal sections are preferred. Finally, Dextrostixcan be used to obtain an objective measurement oftear sugar level in order to diagnose chronic orbitalCSF leak. If CSF leak is a possibility, the patientshould be referred for neurosurgical consultationprior to any lacrimal surgical intervention.

The authors are grateful to Jack H Dunn, the neurosurgeon involvedin this case.

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Acceptedfor publication 3 December 1985.

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