J. Neurol. Neurosurg. Psychiat., 1968, 31, 214-225 Non-traumatic cerebrospinal fluid rhinorrhoea AYUB K. OMMAYA, GIOVANNI DI CHIRO, MAITLAND BALDWIN, AND J. B. PENNYBACKERI From the Branches of Medical Neurology and of Surgical Neurology, National Institute of Neurological Diseases and Blindness, National Institutes of Health, Public Health Service, United States Department of Health, Education and Welfare, Bethesda, Maryland, U.S.A. There is a certain degree of confusion in the litera- ture concerning nasal leakage of cerebrospinal fluid when trauma is not the cause. The term 'spontaneous cerebrospinal fluid rhinorrhoea' has been used more or less consistently to describe such cases since 1899. Although isolated observations had been made earlier, it was the monograph by St. Clair Thomson (1899) that first clearly described and atttempted to define spontaneous cerebrospinal fluid rhinorrhcea as a clinical entity. Other authors have since essayed to subdivide this condition into 'primary spontaneous' or 'idiopathic' rhinorrhoea when a precipitating cause could not be found and 'secondary spontaneous' rhinorrhcea when a cause, usually a tumour, was discovered (O'Connell, 1964). Quite apart from the nosological adequacy of such a subdivision, the term 'spontaneous' cerebrospinal fluid rhinorrhoea itself would appear to be inexact, bearing no relationship to either the pathogenesis or natural history of the disease. The word 'spontaneous' means 'arising from natural impulse, without external stimulant, or hav- ing a self-contained cause or origin, or arising from or entirely determined by the internal operative or directive forces of the organism' (Shorter Oxford English Dictionary, 1962). It is our thesis that a careful study of the natural history of patients suffer- ing such rhinorrhoea reveals no actual case which can fit such a definition-that is, in being truly 'spon- taneous'. It would therefore be more accurate to describe all such cases, hitherto labelled spontaneous, by the more general term of non-traumatic cerebro- spinal fluid rhinorrhcea. It is then possible to sub- divide this category aetiologically on the basis of facts that emerge from a study of such patients, and assign each case according to the causative lesion. Quite apart from clarifying our understanding, this approach avoids what is essentially a 'waste-basket' category of 'primary spontaneous' rhinorrhcea, for those cases in which the true diagnosis has often not been discovered, as well as dispensing with the logic- 'Present address: Department of Neurosurgery, Radcliffe Infirmary, Oxford. ally impossible phrase 'secondary spontaneous rhinorrhcea'. In an earlier report by one of us (A.K.O.), an attempt was made to provide such a classification of cerebrospinal fluid rhinorrhoea on the basis of an analysis of the literature and the case histories of two patients (Ommaya, 1964). Since then we have been able to collect 18 patients with non-traumatic cerebrospinal fluid rhinorrhoea. Thirteen of these were seen at the Radcliffe Infirmary, Oxford, and five at the National Institute of Neurological Diseases and Blindness, National Institutes of Health, Bethesda, Maryland. For every patient we were able to obtain a report of the current status up to late 1967, constituting a minimum of one year and a maximum of 25 years follow-up. On the basis of this study a unitary hypothesis for non-traumatic rhinorrhoea would appear to be strengthened and the following classification, which is a modification of the one previously published (Ommaya, 1965), is recommended (Table I). It will be made apparent from this aetiological classification that the sub- divisions of rhinorrhoea are closely related in terms of the mechanism of the leakage. The non-traumatic group is subdivided into 'high pressure' and 'normal pressure' categories. This reflects an important difference. In the high pressure category the leakage of cerebrospinal fluid is usually acting as a real safety valve, closure of which will invariably worsen the patient's condition if the causative lesion is not treated. Each of these categories is then subdivided into certain aetiological groups under which the mechanism of the rhinorrheea may be better under- stood. The classification will be analysed in detail in the discussion of this paper where the concept of 'focal atrophy' will also be presented. METHODS The eighteen patients we have studied are briefly des- cribed in the summary of case material shown in Tables II and III. They may be grouped according to their lesions into the two major subdivisions of non-traumatic cerebro- 214 Protected by copyright. on June 8, 2020 by guest. http://jnnp.bmj.com/ J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.31.3.214 on 1 June 1968. Downloaded from
Transcript
J. Neurol. Neurosurg. Psychiat., 1968, 31, 214-225
Non-traumatic cerebrospinal fluid rhinorrhoeaAYUB K. OMMAYA, GIOVANNI DI CHIRO, MAITLAND BALDWIN, AND
J. B. PENNYBACKERI
From the Branches ofMedical Neurology and of Surgical Neurology, National Institute ofNeurologicalDiseases and Blindness, National Institutes ofHealth, Public Health Service, United States Department of
Health, Education and Welfare, Bethesda, Maryland, U.S.A.
There is a certain degree of confusion in the litera-ture concerning nasal leakage of cerebrospinal fluidwhen trauma is not the cause. The term 'spontaneouscerebrospinal fluid rhinorrhoea' has been used more orless consistently to describe such cases since 1899.Although isolated observations had been madeearlier, it was the monograph by St. Clair Thomson(1899) that first clearly described and atttempted todefine spontaneous cerebrospinal fluid rhinorrhcea asa clinical entity. Other authors have since essayed tosubdivide this condition into 'primary spontaneous'or 'idiopathic' rhinorrhoea when a precipitating causecould not be found and 'secondary spontaneous'rhinorrhcea when a cause, usually a tumour, wasdiscovered (O'Connell, 1964). Quite apart from thenosological adequacy of such a subdivision, the term'spontaneous' cerebrospinal fluid rhinorrhoea itselfwould appear to be inexact, bearing no relationshipto either the pathogenesis or natural history of thedisease. The word 'spontaneous' means 'arising fromnatural impulse, without external stimulant, or hav-ing a self-contained cause or origin, or arising fromor entirely determined by the internal operative ordirective forces of the organism' (Shorter OxfordEnglish Dictionary, 1962). It is our thesis that acareful study of the natural history of patients suffer-ing such rhinorrhoea reveals no actual case which canfit such a definition-that is, in being truly 'spon-taneous'. It would therefore be more accurate todescribe all such cases, hitherto labelled spontaneous,by the more general term of non-traumatic cerebro-spinal fluid rhinorrhcea. It is then possible to sub-divide this category aetiologically on the basis offacts that emerge from a study of such patients, andassign each case according to the causative lesion.Quite apart from clarifying our understanding, thisapproach avoids what is essentially a 'waste-basket'category of 'primary spontaneous' rhinorrhcea, forthose cases in which the true diagnosis has often notbeen discovered, as well as dispensing with the logic-'Present address: Department of Neurosurgery, Radcliffe Infirmary,Oxford.
In an earlier report by one of us (A.K.O.), anattempt was made to provide such a classification ofcerebrospinal fluid rhinorrhoea on the basis of ananalysis of the literature and the case histories of twopatients (Ommaya, 1964). Since then we have beenable to collect 18 patients with non-traumaticcerebrospinal fluid rhinorrhoea. Thirteen of thesewere seen at the Radcliffe Infirmary, Oxford, andfive at the National Institute of Neurological Diseasesand Blindness, National Institutes of Health,Bethesda, Maryland. For every patient we were ableto obtain a report of the current status up to late1967, constituting a minimum of one year and amaximum of 25 years follow-up. On the basis of thisstudy a unitary hypothesis for non-traumaticrhinorrhoea would appear to be strengthened and thefollowing classification, which is a modification ofthe one previously published (Ommaya, 1965), isrecommended (Table I). It will be made apparentfrom this aetiological classification that the sub-divisions of rhinorrhoea are closely related in termsof the mechanism of the leakage. The non-traumaticgroup is subdivided into 'high pressure' and 'normalpressure' categories. This reflects an importantdifference. In the high pressure category the leakageof cerebrospinal fluid is usually acting as a real safetyvalve, closure of which will invariably worsen thepatient's condition if the causative lesion is nottreated. Each of these categories is then subdividedinto certain aetiological groups under which themechanism of the rhinorrheea may be better under-stood. The classification will be analysed in detail inthe discussion of this paper where the concept of'focal atrophy' will also be presented.
METHODS
The eighteen patients we have studied are briefly des-cribed in the summary of case material shown in Tables IIand III. They may be grouped according to their lesionsinto the two major subdivisions of non-traumatic cerebro-
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Table II lists the patients with normal pressure leaks inwhom corrective surgery for the lesion and for the leakwas usually conducted as one operation. Six out of eightcases (75 %) were cured by surgery. Operative mortalitywas nil and morbidity was confined to one patient. In
Table III, the important clinical data of 10 patients withhigh pressure leaks are presented. In five of these patients,surgical treatment was aimed at the causative lesion andnot at the fistula itself; this approach resulted in stoppageof the rhinorrhaea in four of these patients. In four out of10 cases surgery was also directed at the leak, but in onlyone of these cases could the leak be stopped. In one casethe surgical treatment was neither for the removal of thelesion nor the closure of the fistula but consisted of by-passing an obstruction in the ventricular system with aTorkildsen shunt. This cured the rhinorrhcea.
Illustrative histories of 10 patients selected to demon-strate important points of diagnosis and treatment willnow be presented. This will be followed by a discussion ofthe mechanisms, diagnosis, and management of non-traumatic cerebrospinal fluid rhinorrhoa in the light of acritical review of the literature and our own experience.
Ayub K. Ommaya, Giovanni Di Chiro, Maitland Baldwin, and J. B. Pennybacker
CASE REPORTS
CASE 1 (J.Y. (N.I.H.) No. 7 in Table II). A 48-year-oldmale physicist was quite well until the autumn of 1965when a sudden onset of leakage of cerebrospinal fluidfrom the left nostril was noted. Initially scanty and inter-mittent, the rhinorrheea soon became copious. Physicalexamination was negative. Plain radiographs of the skulland detailed tomograms of the sellar region showed thatair and fluid were present in the sphenoidal sinus. Withthe patient in supine position and the x-ray beam hori-zontal (brow-up view) only a little amount of air was seenin the anterior part of the sphenoidal sinus with a clearair-fluid level also being demonstrated (Fig. la). When thepatient was turned to the prone position, a large spurt offluid came out from the left nostril and the lateral (brow-down) radiograph (Fig. lb) showed that the sphen-oidal sinus was almost in its entirety air filled. The air-fluid level was no longer present. Sellar tomogramsdemonstrated that a break of continuity-an actual hole-was present on the left of the midline immediately behindthe planum sphenoidale (Fig. 2c). Pneumoencephalo-graphy revealed only slight enlargement of the ventricularsystem with no abnormalities in the sellar region; inparticular, no air penetrated within the boundaries of thesella turcica. RISA-cisternography showed an abnormalcollection of activity in the sphenoidal sinus with thepatient supine, confirming that the leak was from thesella turcica region (Figs. 2a, b). When the patient wasput in the prone position, a spurt of fluid-which proved
to be very 'hot' in the well counter-came out from theleft nostril. Lateral scans with the patient replaced in theprone position now revealed that the abnormal sphenoid-al collection of radioactivity could no longer be demon-strated (Fig. 2c). It appeared clear, therefore, that the siteof the leak was into the sphenoidal sinus, probably fromthe 'hole' seen just in front of the sella turcica on the leftside. Surgical exploration by a bi-frontal intraduralapproach was then conducted. The olfactory grooveswere deeper than usual and the arachnoid covered ol-factory bulbs did not fill the anterior part of the cribiformplate completely. This was a bilateral phenomena butthere appeared to be no holes here. The planum sphen-oidale and the remainder of the anterior cranial fossa werequite normal. However, just posterior to the limbussphenoidalis, immediately on the left of the midline, adefinite hole could be palpated. A blunt dissector couldpass without effort through this hole leading into theregion of the sphenoid sinus. Accordingly, a crushedmuscle plug was made to fill this hole snugly and coveredwith a further plug of gelfoam. A patch of gelfoam wasalso left intradurally over the left cribiform plate wherethe left olfactory tract had been deliberately dividedduring the exposure. Post-operatively the patient madean uneventful recovery and had no leakage of cerebro-spinal fluid. Repeated RISA cisternography one monthafter surgery showed no evidence of isotope leakage intothe sphenoid sinus or nasal area. Six months after surgery,the patient is well and has had no further leakage ofcerebrospinal fluid.
TABLE IIISUMMARY OF CASE MATERIAL
B. HIGH PRESSURE LEAKS
Surgery for Surgery for Result for Result Jbrlesion rhinorrhtea lesion rhinorrhaa
No. Lesion
9 Pituitary adenoma. Leak via Yes (1)sellar floorN.W.
10 Acoustic neurinoma. Leak via Yes (1)eustachian tube?E.K.Acoustic neurinoma. Leak via Yes (2)eustachian tube?J.D.
12 Nasopharyngeal carcinoma Yes (1)A.H.
13 Pituitary adenoma Yes (1)E.R.
14 Pituitary tumour No DXr
Q.J.
15 Pituitary adeno-carcinoma.Leak via sellar floorF.K.
Yes (2)
Yes (2)
No
No
No
+DXr No
:ronly No
+DXr Yes
16 3rd ventricle tumour. Leak in Nocribiform plateP.M.
17 Cerebellar glioma. Leak via Yes (3)cribiform plateB.L.
18 Cerebellar glioma. Leak via Yes (3)cribiform plateN.L.
Numbers in parentheses refer to number of operations.DXr = Deep x-ray therapy.
Cured
Cured
Cured
Recurred Leak persists 6 years aftersurgery.On replacement therapyand penicillin
Cured No leak for 6 years
Cured
Persisted Stopped
Persists
? Cured
? Cured
Yes (Shunt) Persists
Yes (1)
Yes (2)
? Cured
? Cured
Stopped
Persists
Cured
Cured
Cured
No leak for 9 years
Died 1 year. No P.M.
No leak 5 years later
Minimal leak persists.Patient refuses surgery6 yearsMassive tumour regressedafter radiation. No tumourrecurrence for 3 years.Leak arrestedNo leak 11 years
No leak or tumour re-currence 6 years
Persisted Died after 2nd leak surgeryfrom pulmonary embolism
Follow-up results
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FIG. 1. Plain skull tomograms. (a) 'Brow-up' view: airbubble in sphenoidal sinus with air-fluid level (arrow). (b)'Brow-down' view: sphenoidal sinus almost completely airfilled. (c) Bone defect (arrow). Patient No. 7 (Table II).
FIG. 2. (a) Leakage and accumulation ofradioactive tracerin sphenoidal sinus (arrows) in two-hour RISA-cisterno-gram. Patient No. 7 (Table II). (b) Normal two-hour RISA-cisternogram in another patient for comparison.
FIG. 2c. Normal post-surgery RISA-cisternogram. Com-pare with pre-surgery in Figure 2a.
Ayub K. Ommaya, Giovanni Di Chiro, Maitland Baldwin, and J. B. Pennybacker
Comment Surgical exploration confirmed the radio-graphic finding of a bony defect in front of the sellaturcica. The nature of this lesion was such that either acongenital or an 'atrophic' aetiology was possible.
CASE 2. (K.P. (R.I.) No. 5 in Table II). A 24-year-oldman had had intermittent leakage of cerebrospinal fluidfrom the left nostril for six years before his admission forinvestigation in 1956. Three attacks of meningitis (onemeningococcal and two pneumococcal) had also occurred.There was no history of trauma or other obvious cause forthe rhinorrhcea. Examination and radiological investiga-tion gave entirely normal results. No contrast studies orattempts to trace the pathway of the leak were made. InAugust 1956 a left frontal intradural exploration was doneand revealed a 2 x 3 mm hole in the postero-lateralcorner of the left cribiform plate, through which a slendercerebral and meningeal hemia entered the nasal cavity.There were no other abnormalities. The hernia wassevered and the dural hole repaired with a pericranialpatch placed intradurally. No attempt was made to closethe bony defect. The patient made an uneventful recoveryand had no further leakage. He was completely well at theend of 1965-that is, nine years after surgical treatment.Comment This patient's course illustrates the ease
with which a probable congenital anomaly responsiblefor a normal pressure cerebrospinal fluid leakage can betreated. That this is not always so is demonstrated by thenext patient.
CASE 3 (B.B. (R.I.) No. 6 in Table II). A 52-year-oldfemale was first seen in 1952 with a short history of thesudden onset of cerebrospinal fluid rhinorrhoea from theleft nostril in October 1951. There was no history oftrauma or other predisposing factors. In January 1952 sheunderwent a left frontal craniotomy by Mr. W. McKissockwhich revealed a small dural hole anterior to the leftolfactory bulb in what seemed to be an excavated cribi-form plate. This was plugged with muscle and oxycel.Post-operatively the leakage recurred and persisted. Threefurther operations were done, in February, March, andJune of 1952, all being left frontal craniotomies, andattempts to plug the same hole with muscle, oxycel, andfascia were repeated on each occasion. The fourth crani-otomy necessitated amputation of the left frontal pole. Inspite of repeated surgery the cerebrospinal fluid rhinor-rhcea persisted and indeed became very profuse in January1953. During this year of continuing rhinorrhoea she alsogained over 50 lb. in weight. At this time she was admittedto the Radcliffe Infirmary and examination revealed anobese woman with a continuous leakage of cerebrospinalfluid from the left nostril (10 to 14 drops/min). She waseuphoric and lethargic. Radiographs of the skull andpneumoencephalograms were interpreted as beingnormal. In October 1963 she underwent bifrontal crani-otomy and intradural exploration of both frontal fossae.The dura over the posterior part of the right olfactorygroove had a 2 x 5 mm hole to which the brain wasadherent. On the left side, no abnormalities were seen buta probe placed in the dural hole on the right passed intothe left posterior ethmoidal sinus. Accordingly, the holeon the right was plugged with muscle and an intradural
fascial repair of the dural defect was completed. Post-operatively she had immediate cessation of therhinorrhoea.Ten days later the cerebrospinal leakage recurred andcontinued unremittingly thence onward. Ten years aftersurgery (February 1963), while in bed with asthma andbronchitis, she died quite suddenly having no remission ofher rhinorrhoea. Necropsyrevealed dense adhesions of themeninges to the base of the skull in the left anterior andmiddle fossae. Unfortunately, an extensive study of theskull and brain for the anatomy of the leakage was notmade, but there appeared to be no evidence for an intra-cranial tumour or other precipitating cause for therhinorrhoea. There was no fracture present and theadhesions in the left-middle fossa were thought to connectwith the sphenoid sinus.Comment This patient demonstrates the extreme
difficulties which may arise in some cases when the exactpathway for the leakage is not precisely visualized at thevery onset. The continuation of the leakage from the leftnostril, in spite of extensive and thorough repair withinthe anterior cranial fossa, coupled with the operativefinding of a track into the left ethmoidal region and theautopsy finding of adhesions in the left middle cranialfossa, raise the possibility of a sellar or parasellar channelinto the sphenoid sinus which was never disclosed.
CASE 4 (C.S. (R.I.) No. 1 in Table II). A 54-year-oldfemale suddenly developed a cerebrospinal fluid leakagefrom the left nostril; there was no prior history oftraumaor other predisposing factors. Two months later shebecame very ill for one month with pneumoccal meningitisfollowing which the rhinorrhoea slowly decreased andfinally ceased altogether six months after its onset. Sheremained well for five years and then once more theleakage of cerebrospinal fluid began. This second siege ofrhinorrhoea was followed by a second bout of meningitis.This time the leakage did not cease but persisted in anintermittent fashion. Neurological examination at theRadcliffe Infirmary was entirely normal and routineradiographs of the skull showed no visible bony defects.A left frontal craniotomy was done in May 1956. Intra-dural exploration of the left anterior cranial fossa revealeda vascularized sleeve of arachnoid passing through a smallhole in the left cribiform plate. A piece of temporal fasciawas laid intradurally over the entire left cribiform plateregion to seal off the defect after dividing the arachnoidsleeve. Post-operatively the patient made an uneventfulrecovery and there was no evidence of cerebrospinal fluidrhinorrhcea. However, intermittent leakage recurred aftera few months and persists to this date. The patient has notdesired further surgery and has not had any meningitis.Comment This was an apparently simple case of
normal pressure cerebrospinal fluid leakage through thecribiform plate. Recurrence of the rhinorrhoea occurredafter temporary post-operative cessation. Either the intra-dural fascial graft was inadequate to seal the fistula orthere was another fistula which was not diagnosed.
CASE 5 (R.F.J. (N.I.H.) No. 2 in Table II). A 57-year-oldmale was quite well until 1958 when a transient episode ofsudden loss of consciousness occurred at work. One yearlater a similar brief episode and nocturnal tongue biting
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necessitated anti-convulsant therapy. No further tonguebiting or unconsciousness was noted. In March of 1963sudden leakage of clear fluid from the left nostril began.He was admitted to the National Institutes of Health,Branch of Surgical Neurology, in July 1963. Physicalexamination revealed no other abnormality of note.Several encephalograms confirmed a left fronto-temporalepileptogenic focus. Endocrinological investigationsrevealed mild hypopituitarism. Plain radiographs of theskull and detail tomograms showed an enlargement of thesella turcica. At pneumoencephalography, air filled theanterior half of the sella turcica (Fig. 3). The widenedcisterna intrasellaris was thus causing an 'empty' sellaturcica. Bilateral carotid angiography revealed noabnormality. RISA-cisternography showed that the leakof cerebrospinal fluid was from the antero-inferior part ofthe sella turcica exactly corresponding with the arach-noidal 'hernia' into the 'empty sella' seen at pneumoence-phalography (Fig. 4a). A right frontal craniotomywas then performed and an intradural approach to thesella revealed the arachnoidal sleeve entering the sella.This was incised and with a dissector one could feel theempty anterior space plus tissue in the posterior sella.Several pieces of gelfoam were then placed into the emptyanterior arachnoid pouch so as to plug it entirely. Thepatient made an uneventful recovery and, in spite of blow-ing his nose in the post-operative period (against firminstructions to the contrary), no further cerebrospinal fllidrhinorrhcea was noted. Six months after this operation liewas re-admitted for a check RISA cisternography whichnow proved to be normal (Fig. 4b). Since that time thepatient has continued to do well, and when last seen-that is, over three years after surgery-there was norecurrence of cerebrospinal fluid rhinorrhcea or othersignificant disorder.
ComZ?menit This is an example of normal pressure non-traumatic cerebrospinal fluid rhinorrhoea occurring in apatient presenting initially with epilepsy of late onset.
Suspicion of an intracranial tumour was increased whenthe rhinorrhcea began and the enlarged sella suggested apituitary tumour. However, pneumoencephalographyrevealed the arachnoid 'hernia' into an empty sella. Thefistula was clearly demonstrated by RISA cisternographyand surgical treatment was successful after closure of thesellar hole with gelfoam only. A similar lesion was foundand treated in patient No. 8 in Table II and an operativeview of her sellar region can be seen in Fig. 5.
CASE 6 (N.W. (R.I.) No. 9 in Table 111). A 44-year-oldwoman began to suffer episodes of psychomotor epilepsywith evidence of mild hypopituitary disturbance in 1957.Examination revealed a right homonymous hemianopsiaand a left 3rd nerve palsy. A clinical diagnosis of pituitaryadenoma was made and radiotherapy plus hormonereplacement resulted in excellent recovery. Three yearslater she noted the sudden onset of cerebrospinal fluidrhinorrhoea from the left nostril. After 10 weeks and twomild attacks of meningitis she was admitted to hospital.Examination revealed a minimal right quadrantanopsiaand left carotid angiogram showed extrasellar extensionof the pituitary tumour. A left frontal craniotomy allowedtotal removal of the tumour, and a muscle plug was placedin the cavity created within the sella turcica. During thisoperation no fistula was found. Post-operatively thepatient had no further rhinorrhaea for six months. In late1960, the leakage recurred from the left nostril with noother evidence of tumour recurrence. A second frontalcraniotomy showed the sella to be almost empty, and aprobe passed easily into the sphenoid sinus. The sella waspacked with muscle and a free pericranial graft coveredthis area as well as the left anterior cranial fossa. Nofurther cerebrospinal fluid rhinorrhoea was noted untilMay 1963-that is, three years later. Her physicianselected on this occasion to place her on long-term anti-biotics without further surgery. At the present time shecontinues to have cerebrospinal fluid rhinorrhaea but has
- =a
FIG. 4a, b. Samtie patientt as Fig. 3. RISA-cisternogramn showvingfinidintgs after slurgical clure (b).
leakage ol' tracer from sella legiont (a). Normz1a
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had no further attacks of meningitis while continuingon penicillin and hormone replacement therapy. Thevisual field defect has not increased.Comment In this case the cerebrospinal fluid leakage
followed a course of deep x-ray treatment directed at alarge pituitary adenoma. It is possible that radiation-induced tumour atrophy resulted in a cystic intrasellarextension of the cerebrospinal fluid space which in turnproduced the fistula through a portion of the sellar flooreroded by the tumour's growth.
CASE 7 (J.D. (R.I.) No. 11 in Table III). A 34-year-oldman was first seen in the E.N.T. Department in July 1955with a history of deafness in the left ear since childhood,a left facial palsy of 11 years' duration that had improvedpartially, and profuse cerebrospinal fluid rhinorrhoa forone year. Examination revealed that in addition to theleft 7th and 8th nerve lesions there was bilateral nystag-mus. Radiographs of the skull revealed an eroded apex ofthe left petrous temporal bone. An E.N.T. surgeonexplored the petrous area via a mastoid approach andwas able to uncover and partially excise an acousticneurinoma on this side. Post-operatively the cerebrospinalfluid rhinorrhoea continued unabated. In March 1957,after a severe attack of meningitis, he was admitted to theNeurosurgical Department. A posterior-fossa explorationrevealed a large left acoustic neurinoma which was fillingthe cerebello-pontine angle as well as eroding into thepetrous apex. This was removed subtotally. The patientmade a good recovery and no further leakage was noted.Nine years after this surgery, the patient had no recurrenceof either the tumour or the rhinorrhoea.Comment This was a slowly growing intra- and extra-
petrous acoustic neurinoma which precipitated a cerebro-spinal fluid rhinorrhcea either by the high pressureproducing a leak through the cribiform plate or byestablishing a fistula into the Eustachian tube via themiddle ear cavity. Unfortunately the exact fistula couldnot be demonstrated. An otological approach to thepetrous apex revealed, and allowed biopsy of, the tumour,but the leakage was not reduced after this type of surgery.However after a posterior-fossa intracapsular removal ofthe tumour without attempting to repair mhe fistula, therhinorrhoea stopped. Posterior fossa surgery cured the
rhinorrhrea either by pressure reduction, closure of thefistula by adhesions, or both of these factors.
CASE 8 (B.L. (R.I.) No. 17 in Table III). A 28-year-oldman presented in 1959 with bouts of cerebrospinal fluidrhinorrhoea and occasional bi-occipital headaches. Thesignificant past history included three operations for aright cerebellar astrocytoma-an aspiration biopsy in1936, a subtotal excision in 1937, and a total removal in1948. From then on he had no complaints until the onsetof the rhinorrheea in 1959. This continued with varyingintensity until 1960 when he was admitted for treatment.The leakage could be easily started by tipping his headforward. Examination revealed no bulge of the posteriorfossa craniectomy site, no papilloedema, and minimalneurological deficits related to the cerebellar tumourwhich were unchanged from that noted in 1948. Radio-graphs of the skull showed marked osteoporosis of thewalls of the sella and a large midline bony defect anteriorto the tuberculum sellae. A lumbar puncture (whilerhinorrhnea was present) showed cerebrospinal fluid at apressure of 120 mm and with 160 mg of protein. Panto-paque ventriculography with 2-5 ml ofdyewasthen carriedout via the right frontal horn, and the dye flowed easilythrough the 3rd ventricle, aqueduct, and 4th ventricle intothe basal cisterns. No evidence of a space occupying lesionin the posterior fossa was found. A bifrontal craniotomywas then performed and intradural exploration revealedmultiple meningo-cerebral herniae into bony defects closeto the midline in the anterior cranial fossae. Fascia latagrafts were used bilaterally to cover the holes after theherniae were excised. For a few days post-operativelythere was a moderate amount of blood-stained rhinor-rhoea but this rapidly decreased by the end of the firstweek. At discharge two weeks later, there was no rhinor-rhoea. The latest follow-up in 1966 (six years after surgeryfor rhinorrhoea and 30 years after tumour diagnosis)revealed the patient to be in excellent health with norecurrence of either rhinorrhcea or cerebellar tumour.Comment This patient's rhinorrhoea was a delayed
consequence of the posterior fossa tumour. The bonyabnormalities and brain + meningeal herniae wereprobably initiated by high pressure, but the leakage didnot start for 11 years after radical removal of the tumour.The mechanism for delayed leakage in this case may berelated to that suggested for Case 6 (N.W.).
CASE 9 (P.M. (R.I.) No. 16 in Table III). A young girlbegan to suffer severe headaches and blurred vision in1934. In 1936 a surgeon in London performed bitemporaldecompressive craniectomies following which all hersymptoms vanished. She remained quite well until 1939when she developed a left hemiplegia and recurrence ofthe headaches. This was presumed to be due to a rightcerebral hernia at the site of decompression. Three yearslater in 1942 she suddenly began to leak cerebrospinalfluid from her nose, and this rhinorrhoea was associatedwith a dramatic improvement in her headaches, hemi-plegia, and general well being. She remained well but withthe fistula leaking until 1945 when the leakage suddenlystopped. She immediately became hemiplegic once moreon the left and the headaches recurred. Examination at
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this time was made in the Neurosurgical Department ofthe Radcliffe Infirmary. This revealed bulging in bothareas of subtemporal decompression, mild secondary opticatrophy, and a moderate left hemiparesis. Routineradiographs of the skull revealed a large suprasellarcalcification forming an almost complete cast of the 3rdventricle, including an extension into the right foramen ofMonro. The diagnosis of a calcified craniopharyngiomawas made and in May 1945 a Torkildsen shunt was carriedout. The pressure in the right lateral ventricle at this timewas 250 mm cerebrospinal fluid. Post-operatively shemade an uneventful recovery and had no further rhinor-rhea although the left hemiparesis persisted. When lastseen, almost 20 years after the Torkildsen shunt wasplaced, the patient was in good health with no headachesor rhinorrhoa, but with a persisting left hemiparesis.Comment This unusual case is an excellent example of
the 'safety-valve' nature of a high-pressure non-traumaticcerebrospinal fluid rhinorrhoea. After the tumour was by-passed by a Torkildsen shunt, the rhinorrhcea remained inabeyance and has not recurred over a period exceeding 20years.
CASE 10 (N.L. (R.I.) No. 18 in Table 111). A 10-year-oldboy began to have episodes of headache, vomiting, andataxia in 1933 leading to a posterior fossa decompressionin 1939. Further deterioration in 1944 led to re-explorationand sub-total removal of a pilocytic cerebellar astrocy-toma by Sir Hugh Cairns. He made an excellent recovery,being able to return to work on his farm and remainedwell until 1955. At this time the sudden onset of cerebro-spinal fluid rhinorrhaea from the right nostril was noted.He was readmnitted and a right frontal craniotomyrevealed a cerebrospinal fluid fistula through the cribi-form plate on the right side which was covered with afascia lata graft placed intradurally. An uneventfulrecovery with no further rhinorrhcea after this operationended one month later with signs of raised intracranialpressure. However, these signs subsided in a few weeksand the patient returned to work. He was fairly well forabout two years with only one 10-day spell of cerebro-spinal fluid rhinorrhoea in 1956. In 1957 the rhinorrhceastarted again and was followed by a persistent low-grademeningitis. After this was controlled he underwentexploration of the posterior fossa. No evidence of tumourcould be found. Post-operatively meningitis recurred andrequired further intensive therapy. Approximately twomonths after the latest operation his condition improvedbut rhinorrheta persisted and accordingly the right frontalcraniotomy was re-opened. About one large and threesmall brain and meningeal herniae were seen passing intoholes in the right anterior cranial fossa just lateral to thecribiform plate. The herniae were sectioned and a fascialgraft laid over the meningeal defects. In addition ananterior third ventriculostomy was performed. After thisoperation, however, the rhinorrhoea persisted and therewas a considerable accumulation of intracranial air in thefrontal region. Therefore, a further craniotomy was per-formed and a plug of acrylic placed in the largest of thebony defects external to the fascial graft which was foundto be thoroughly adherent to the dura mater. Post-operatively his level of consciousness did not improve
and the posterior fossa defect was noted to be bulging. Nofurther rhinorrhcea occurred but his general conditionworsened as the chronic meningitis reasserted itself inspite of intensive systemic and intrathecal antibiotictherapy. About two months later, in January 1958, hesuddenly collapsed and died. Necropsy revealed the causeof death to be thrombosis of the inferior vena cava as wellas pulmonary emboli. There was no evidence of tumourrecurrence and the frontal cerebrospinal fluid fistulaewere well closed.Comment This case is a further illustration of the
'safety valve' nature of some cerebrospinal fluid rhinor-rhceas. The persistence of meningitis in this patientcomplicated the management severely and renderedrepeated efforts to stop the leak ineffective until too late.It is interesting to note that 'pressure' phenomenarecurred when the leak stopped, in spite of the absence oftumour recurrence. This may have been due to a degreeof communicating hydrocephalus.
DISCUSSION
The case-histories presented were chosen to illustratefacets in the pathogenesis as well as diagnosis andtreatment of non-traumatic cerebrospinal fluidrhinorrheoa. Before reviewing these aspects of theproblem in detail, a clarification concerning ourclassification must be presented. The aetiologicalclasses shown in Table I are not mutually exclusivein so far as the mechanics of the cerebrospinal fluidleakage itself are concerned. That is to say, thesecategories overlap at all levels including the firstmajor subdivision into traumatic and non-traumaticrhinorrhoeas. Thus it is possible that in certain casesof delayed traumatic cerebrospinal fluid leakage theactual precipitating cause for the rhinorrhaea may beanalogous to mechanisms producing non-traumaticleaks. This would explain case reports of patientswith delayed traumatic cerebrospinal rhinorrheea inwhom associated congenital anomalies related to thefistula are found-for example, persistence ofthe basi-occipital canal (Voena, 1959). Similarly theonset of cerebrospinal fluid rhinorrheea in certaincases of pituitary tumour treated with x-ray alonesuggests a similarity between post-radiation atrophyand the post-traumatic atrophy of cerebral tissueswhich may be responsible for the onset of delayedtraumatic cerebrospinal fluid rhinorrhoea (Bateman,1966).
PATHOGENESIS A previous paper by one of us(A.K.O.) contains a full review of earlier reportspublished up to 1964 (Ommaya, 1964). Accordingly,only more recent papers will be referred to in thefollowing discussion. The category of cerebrospinalfluid rhinorrhoea caused by tumours is subdividedinto 'direct' and 'indirect' in Table I. This indicatesthe two ways in which the fistula may be created by
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Ayub K. Ommaya, Giovanni Di Chiro, Maitland Baldwin, and J. B. Pennybacker
the tumour-that is, either directly by erosion ofmeninges and bone (Cases 9 through 15 in Table III)or indirectly via pressure erosion of anatomicallyfragile areas of the skull base (Cases 16, 17, and 18 inTable III). It is significant that in all three of theseindirect rhinorrhoeas the fistula was in the cribiformplate; an indication of the fragility of this area.Previous reports on tumour-induced cerebrospinalfluid rhinorrhcea (Teng and Papatheodorou, 1965;Raskind, 1965; Gotham, Meyer, Gilroy, and Bauer,1965) have emphasized that pituitary tumours are thecommonest lesion and in our series of 10 patientswith high pressure leaks there were four cases (Cases9, 13, 14, and 15 in Table III). It should be stressedthat cerebrospinal fluid rhinorrhoea is an extremelyrare complication of such tumours.
Hydrocephalus This is a much rarer cause ofcerebrospinal fluid rhinorrhoea. The earliest report ofa case due to communicating hydrocephalus is byMiller and little can be added to that account (Miller,1826). Occasionally hydrocephalus is associated witha congenital anomaly as the cause of a high pressurecerebrospinal fluid leak and such an association hasbeen reported with Crouzon's disease and Albers-Schonberg disease (osteopetrosis). In the majority ofcases, however, congenital anomalies are aetiologic-ally more important for normal pressure leakages.Here nasal encephalocele is the usual lesion dis-covered (Case 4 in Table II) and such patients are theonly examples of normal pressure leakages occurringin childhood.Focal atrophy Congenital anomalies may also
play a role in the next category of our classification,namely 'focal atrophy'. This is a speculative ideasuggesting that the noimal contents of the cribiformplate or sella turcica areas are reduced in bulk. Thisatrophy may be due to ischemia. The empty spacethus created is filled with a cerebrospinal fluid ladenarachnoidal pouch which is an enlargement of thenormal arachnoidal space extensions. This pouchenables the normal cerebrospinal fluid pressure pulseto exert a focal and continually erosive effectanalogous to the creation of cranial vault excavationsby the arachnoidal granulations. This concept offocal atrophy was developed to explain the mechan-ism of cerebrospinal fluid rhinorrheea in three of ourpatients with sellar leaks (Cases 2, 7 and 8 in TableII). But it may be invoked to explain normal pressurecerebrospinal fluid leakage from any site in adults.Thus it agrees quite well with the hypothesis ad-vanced by O'Connell. He described in two patients acombination of an excavated lamina cribrosa plus ashrunken olfactory bulb enabling the development ofa pulsating pocket of cerebrospinal fluid over thefistula (O'Connell, 1964). In our series, Case 1 inTable II appeared to have a similar lesion.
Minor degrees of congenital meningeal andmeningo-cerebral herniae can also act as actual orpotential pathways in the anterior skull base andpresent in adult life with cerebrospinal fluid rhinor-rhoea-for example, Cases 3, 5, and 6 in Table II.The majority of case reports of'primary spontaneous'rhinorrheea are explicable by this aetiology. Thegenesis of such a leak, aside from the pole of focalatrophy, is probably related to the attainment of themaximum cerebrospinal fluid pressure in adults,which is almost three times that of infants, as well aswith factors such as sneezing, coughing, or othercauses of normal fluctuations in cerebrospinal fluidpressure (O'Connell, 1953).
Osteomyeletic erosion The final aetiological cate-gory is of osteomyeletic erosion. This is a very rarecause (Nori and Carteri, 1964) and we have not had asingle patient with this condition.
DIAGNOSIS This includes identification of cerebro-spinal fluid as the constituent of the rhinorrhoea,demonstration of the cause whenever possible, as wellas localization of the fistula itself. Identification ofcerebrospinal fluid is usually easy except in a fewcases of 'vasomotor rhinorrhoea' or allergic rhinitiswhere a faulty initial diagnosis can be misleading.However, persistence of a clear non-sticky nasaldischarge should immediately rouse suspicions andlead to further investigations. Biochemical tests onthe collected fluid, particularly the often quoted useof glucose-oxidase test papers, are not fully reliable.The latter method has been shown to have a 45 to75% chance of positive results with normal nasalsecretions (Gadeholt, 1964). Positive identification ofcerebrospinal fluid necessitates introduction of suit-able tracers into the cerebrospinal fluid cavities andtheir recovery in the nasal discharge. These tech-niques are part of fistula localization and will beconsidered below.
Concerning demonstration of the cause of theleakage it should be remembered that over half thecases of non-traumatic rhinorrhoea are high pressureleaks due to tumours, and the diagnostic approach tosuch patients should therefore be the same as that topatients with epilepsy of late onset. However, it is notenough to display the causative space-taking lesion;the fistula must also be localized. Naturally suchlocalization takes precedence when a normal pressureleak is present.
It cannot be sufficiently emphasized that theassessment of the location of the leakage is often adifficult and always a challenging diagnostic problem.This is due to the fact that the possible leaking sitesare quite numerous and may occur in the anterior,middle, and posterior fossae. Most frequently thecerebrospinal fluid reaches the nasal cavity through
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F ~~223Non-traumatic cerebrospinal fluid rhinorrhuea
the frontal sinus, the lamina cribrosa, the sphenoidalroof, or the petrous bone via the middle ear and theEustachian tube (Fig. 6). The fact that the leak maybe found on the side opposite to the dripping nostriland the possible multiplicity of fistulae furthercomplicate the matter. Considering that if thefistula does not heal spontaneously the patient is inserious danger of recurrent meningitis and that themandatory corrective surgery will be successful onlyif the actual leaking site is exposed at operation, theimportance of a diagnostic localizing method isobvious.To localize the fistula responsible for the cerebro-
spinal fluid loss, one can use dyes, fluorescentsubstances, radiography, or radioactive tracers(Table IV). Dyes (methylene blue, phenolsulfon-phthalein, indigo carmine) have been introducedbefore or during surgery within the subarachnoidalspaces or intranasally forvisual localization ofthe fist-ula. Fluorescein has also been used. Most ofthese dyescan, however, cause significant morbidity (Wolman,1966). Tomography, pneumoencephalography, andsubdural pneumography frequently provide help forthe localization ofthe leaking break. Various attemptshave been made to localize the fistulaby Pantopaqueinjected into the subarachnoidal space (Rockett,Wittenborg, Shillito, and Matson, 1964), as well asthe ventricular system, a pneumocephalic cavity, andintranasally (Teng and Edalatpour, 1963). After theinjection of Pantopaque the passage of the opaquematerial is observed with fluoroscopy and radio-graphy. However, this contrast medium causesmeningeal irritation. In 1956 (Crow, Keogh, andNorthfield, 1956), the use of radioactive sodium
(Na24) was suggested. This tracer was injected intothe cisterna magna and cotton pledgets ('patties'),conveniently distributed in the walls and roof of thenose and nasopharynx and upon the openings of theEustachian tubes, were afterwards examined with acounter. The cotton patty showing the highest radio-activity indicated the site of leakage. A similarisotope-counting technique has been reported em-ploying RISA. Isotope-cisternography has been intro-duced by one of us (Di Chiro) and has been usedsuccessfully in many cases of traumatic and non-traumatic cerebrospinal fluid rhinorrhrea. Thetechnique and results have been previously reported(Di Chiro, Reames, and Matthews, 1964; Di Chiroand Grove, 1966; Ommaya, 1966). In Table V thetracers used for isotope-cisternography and ventricu-lography are listed. Limiting ourselves to non-traumatic cerebrospinal fluid rhinorrhcea, we feelthat a combination of accurate radiographic studiesand isotope-cisternography give the best results.Plain radiographs may demonstrate air-fluid levelswithin the sphenoidal sinus (two of our cases) as wellas reveal an enlarged sella turcica. With tomographya bone defect may be demonstrated (one ofour cases).At pneumoencephalography a dilated intrasellarsubarachnoid space may be put in evidence (two ofour cases). The intrasellar subarachnoid pocket(Busch, 1951; Engels, 1958), acting as a tense pulsat-ing cyst, is probably responsible for a break in thesellar floor. The pneumoencephalogram is essentialto decide between such a cyst and a pituitary adeno-ma. In addition, air studies permitted us in one ofour cases to make the tentative diagnosis of directthird ventricle-cisternal-sphenoidal sinus communi-
FIG. 6. Common pathways ofh cerebrospinal fluid leakage.
I: !!"ILol.
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Intraventricular Within surgical shunts5) 99mTc serum albumin (t")
6) 9-mTc serum albumin with high speicfic activity*Di Chiro: Unpublished data. K
cation (Case 15 in Table III). RISA-cisternographygave, in our hands, the final diagnostic answer in allthe cases (four) in which it was employed. Anothergreat advantage of this technique is the ease withwhich it may be repeated to provide post-operativeand follow-up verification of the efficacy of treat-ment with little or no discomfort to the patient.
TREATMENT This is primarily surgical, althoughnon-surgical measures have resulted in successfulmanagement of such patients on rare occasions.Remission of leakage, rarely permanent, may followlumbar puncture, meningitis, or dye injection. Long-term medical management with antibiotics has beenreported to be possible (Anderson, Schwarz, andGammon, 1961), but with the increasing number ofresistant organisms this conservative therapy becomeshard to justify. Repeated lumbar punctures havebeen tried and recommended but may now beconsidered an obsolete method. Similarly x-raytherapy to the site of the fistula is of no value.Instillation of substances nasally to promote closureof the fistula from below-for example, silvernitrate-has not been successful, but a recent cure of
a cerebrospinal fluid rhinorrheea has been reportedfollowing the application of Eastman 9-10 glue to theroof of the ethmoid sinus (Gotham et al., 1965). It isoften recommended that following the identificationof cerebrospinal fluid in a rhinorrhoea, expectanttreatment, including nursing in a semi-Fowler posi-tion, discouragement of nose blowing, sneezing andstraining, and antibiotic administration, be tried forsix to eight weeks before surgery is considered. Whileit is true that the majority of cases of cerebrospinalfluid rhinorrhcea, particularly the traumatic variety,will stop leaking, it would be risky to suggest thatthis conservative therapy be adopted routinely.Indeed, the occurrence of repeated attacks of menin-gitis after spontaneous closure of such fistulae, evenin the absence of obvious leakage of fluid (Schneiderand Thompson, 1957), would support a more activeapproach to some of these patients. Thus in non-traumatic cerebrospinal fluid rhinorrhoea a conserva-tive attitude is probably justifiable only in thosepatients with high pressure leaks in whom the causallesion cannot be removed. This was the initial man-agement of our Case 15 in Table III. However, afterapparently complete regression of her adeno-carcinoma following deep x-ray therapy, her fistulawas closed intracranially to prevent recurrentmeningitis. In normal pressure leaks surgical repairof the fistula is usually easy and is to be recommendedas soon as the diagnosis is established.
Surgical techniques to repair such fistulae arenumerous but may be considered briefly under twoheads. First, the extracranial, usually transnasal,approach, and, secondly, the intracranial approach.The extracranial, extra-nasal approach to fistulaethrough the frontal and ethmoidal sinuses has beenrecently recommended by Aboulker, Le Beau,Sterkers, and Elbaz (1966). Mennig (1964) hasdescribed a trans-ethmoidal trans-sphenoidal ap-proach to repair a fistula through the floor of thesella turcica which was successful after an intra-cranial attempt at closing the leak had failed. Arecent review of such extracranial techniques is byVrabec and Hallberg (1964). In our opinion, suchextracranial techniques should be reserved for thecarefully selected patient in whom the aetiology ofthe leakage has been clearly understood, the fistulatrack demonstrated, and in whom the intracranialmethods cannot be undertaken for definite reasons.The rationale for this opinion is that cerebrospinalfluid rhinorrhcea is not simply a matter of closure ofthe bony defect but, more importantly, necessitatesrepair of the meningeal defects. Such meningealrepairs are best conducted after adequate inspectionintradurally, and rhinological procedures seldomprovide an adequate view within the dura mater.The intracranial procedures have been adequately
I. 1
II. P
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described (Dandy, 1944) and only a few commentsare necessary. As stated before, the crucial factor inadequate surgical treatment is closure of the menin-geal defect. This is often quite small in non-traumaticleaks and easy to overlook. Some neurosurgeonshave resorted to filling the craniotomy site with salineand with the help of the anaesthesiologist obtain avisual demonstration of the hole by forcing air fromthe nasopharynx into the exposure area where theescape of bubbles may be seen. However, withaccurate pre-operative diagnosis, this manoeuvrewith its attendant risk of sepsis may be avoided.After demonstration of the meningeal defect, thepatient's own fascia, pericranium, muscle, or stripsof gelfoam may be used to provide the intraduralrepair, with fascia being the most satisfactory. It isnot essential to strip the dura and display small bonydefects and attempt to seal them with bone wax,acrylic cements, or other substances. This is necessaryonly where the bony defect is large enough to necessi-tate support of the intradural graft. In such casesmethyl metharylate is a satisfactory caulking material.In summary, the intracranial, intradural approach isrecommended for all non-traumatic cerebrospinalfistulae with careful 'patching' of the fistula site,preferably using the patient's own tissues as freegrafts. Sealing of subjacent bony defects is requiredonly when the hole is of significant size-for example,greater than 2 mm in diameter. In high pressureleaks, removal or by-pass of the tumour or obstruc-tion should always precede repair of the fistula,which should be carried out only where it is positivelyestablished that a 'safety-valve' function is no longerrequired. In normal pressure leaks, obliteration ofthe arachnoid hernia should accompany carefulintradural repair of the fistula. It is hoped thatoptimum management for every case of non-traumatic cerebrospinal fluid rhinorrhoea may beachieved by consideration of these facts.
SUMMARY
We have reviewed our experience with 18 cases ofnon-traumatic cerebrospinal fluid rhinorrhoea. A newclassification eliminating the terms 'primary' and'secondary spontaneous' rhinorrhcea is offered. Twomain categories are recognized: high pressure leaks(10 patients), primarily due to tumours, and normalpressure leaks (eight patients) in which congenitalanomalies and 'focal atrophy' are aetiologicallysignificant. The latter concept is fully explained inthe text. Details of the mechanics of fistula formation,diagnosis, and treatment of such patients are
presented. Particular emphasis is placed on the roleof isotope cisternography in localization of suchcerebrospinal fluid fistulae.
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Anderson, W. M., Schwarz, G. A., and Gammon, G. D. (1961).Chronic spontaneous cerebrospinal rhinorrhea. Arch. intern.Med., 107, 723-731.
Bateman, G. H. (1966). Experiences with cerebrospinal fluid rhinor-rhoea arising from the pituitary fossa. Proc. roy. Soc. Med., 59,169-174.
Busch, W. (1951). Die Morphologie der Sella turcica und ihre Bezie-hungen zur hypophyse. Virchows Arch. path. Anat., 320,437-458.
Crow, H. J., Keogh, C., and Northfield, D. W. C. (1956). The locali-sation of cerebrospinal-fluid fistulae. Lancet, 2, 325-327.
Dandy, W. E. (1944). Treatment of rhinorrhea and otorrhea. Arch.Surg., 49, 75-85.
Di Chiro, G., and Grove, A. S., Jr. (1966). Evaluation of surgical andspontaneous cerebrospinal fluid shunts by isotope scanning. J.Neurosurg., 24, 743-748.Reames, P. M., and Matthews, W. B., Jr. (1964). RISA-ventri-culography and RISA-cisternography. Neurology (Minneap.),14, 185-191.
Engels, E. P. (1958). Roentgenographic demonstration of a hypo-physical subarachnoid space. Amer. J. Roentgenol., 80, 1001-1004.
Gadeholt, H. (1964). The reaction of glucose-oxidase test paper innormal nasal secretion. Acta oto-laryng. (Stockh.), 58, 271-272.
Gotham, J. E., Meyer, J. S., Gilroy, J., and Bauer, R. B. (1965).Observations on cerebrospinal fluid rhinorrhea and pneumo-cephalus. Ann. Otol. (St. Louis), 74, 215-233.
Mennig, H. (1964). Heilung einer schwierigen Liquorfistel durchrhinochirurgisches Vorgehen. Z. Laryng. Rhinol., 43, 412-419.
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O'Connell, J. E. A. (1953). The cerebrospinal fluid pressure as anaetiological factor in the development of lesions affecting thecentral nervous system. Brain, 76, 279-298.
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(1966). Trauma to the nervous system. Hunterian Lecture. Ann.Roy. Coll. Surg. Eng., 39, 317-347.
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Rockett, F. X., Wittenborg, M. H., Shillito, J., Jr., and Matson, D. D.(1964). Pantopaque visualization of a congenital dural defect ofthe internal auditory meatus causing rhinorrhea: Report of acase. Amer. J. Roentgenol., 91, 640-646.
Schneider, R. C., and Thompson, J. M. (1957). Chronic and delayedtraumatic cerebrospinal rhinorrhea as a source of recurrentattacks of meningitis. Ann. Surg., 145, 517-529.
Teng, P., and Edalatpour, N. (1963). Cerebrospinal fluid rhinorrheawith demonstration of cranionasal fistula with Pantopaque.Radiology, 81, 802-806.and Papatheodorou, C. (1965). Cerebrospinal rhinorrhea andotorrhea. Arch. Otolaryng., 82, 56-61.
Thomson, St. C. (1899). The Cerebrospinal Fluid; Its SpontaneousEscape From the Nose. Cassell, London.
Voena, G. (1959). Considerazioni su un caso di grave cranio-rino-liquorrea da presistenza del canale basi-occipitale. Arch. ital.Otol., 70, 212-222.
Vrabec, D. P., and Hallberg, 0. E. (1964). Cerebrospinal fluid rhinor-rhea. Arch. Otolaryng., 80, 218-229.
Wolman, L. (1966). The neuropathological effects resulting from theintrathecal injection of chemical substances. Pararlegia, 4,97-115.
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