T h e Journal o[ P E D I A T R I C S
397
Cerebrospinal fluid otorrhea and recurrent meningitis Eugene B. Spitz, M.D., Seymour Wagner, M.D.,* Joseph Sataloff, M.D., Nicholas P. Hoffman, M.D., and John W. Hope, M.D. PHILADELPI~IA~
PA.
I N F R E Q U ~; N T L Y recurring otitic m e n ingitis resulting from a fracture of the skull, the route of infection m a y be difficult to detect. 1-a This is a report of an unusual case of a child in whom otitic meningitis recurred 20 times, requiring the combined efforts of the pediatric, radiologic, otolaryngologic, and neurosurgical departments of tile Children's Hospital of Philadelphia and 4 surgical procedures before the route of infection was finally discovered and obliterated. From a review of this and similar cases, ~-6 a method of clinical approach is suggested for the investigation of recurrent otitic meningitis.
REPORT OF A CASE This boy had a total of 29 admissions to the Children's Hospital of Philadelphia. He was first admitted in 1948 at 17 months of age with meningitis and bilateral mastoiditis (Table I). T h e meningitis responded well to
From the Children's Hospital o[ Philadelphia, the School o[ Medicine of the University o[ Peitnsylvania, and the Jefferson Medical College, Philadelphia, Pa. -~Address, Jefferson Medical Co~ege and Hospital, 1025 Walnut Street, Philadelphia, Pa.
antibiotic therapy, after which a bilateral simple mastoidectomy was performed. There was no history of trauma to the skull and x-ray examination showed no evidence of any fracture of the skull. During the next year and a half the child had 6 episodes of meningitis. Following tile sixth episode, indigo carmine was injected into the cerebrospinal fluid, but no communication between the ear or nose and the cerebrospinal fluid could be demonstrated. Multiple readmissions for meningitis ensued, all of which were preceded by upper respiratory infections, and on 9 occasions by otitis media. Nine years after the first admission the child was admitted for the sixteenth episode of meningitis. Following recovery, a re-exploration of tile original mastoid cavity on the right side was done, and, again, nothing unusual was noted by the surgeon. On the eighteenth admission injection of dye into the cerebrospinal fluid established tile presence of cerebrospinal fluid rhinorrhea from the right side. Following another bout with meningitis, it became apparent that the child also had cerebrospinal fluid otorrhea in the right ear. T h e otorrhea was confirmed by injecting
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S p i t z et al.
Table I. Summary of admissions and bacteriologic findings Admission
1 2
Diagnosis
Meningitis Meningitis
Throat culture
Nasopharyngeal culture
Cerebrospinal fluid culture
Culture of ear or mastoid
Pneumococcus
Pneumococcus
Staphylococcus
H. influenzae
Staphylococcus
Staphylococcus Pneumococcus
Meningococcus
Negative
Staphylococcus Staphylococcus
Pneumococcus
P n e u m o c o c c u s Pneumococcus H. influenzae
H. influenzae
N. catarrhalis
Pneumocoecus 3
Pharyngitis
N. catarrhalis
Pneumocoecus 4
Meningitis
N. catarrhalis
Pneumococcus 5
Tonsillectomyand adenoidectomy
6
Meningitis
N. catarrhalis
7
8
9
Otitis media
N. catarrhalis
Pneumococcus Streptococcus
Pharyngitis
No growth
Meningitis
N. catarrhalis
Pneumococcus
Pneumococcus Streptococcus
N. catarrhalis
Pneumococcus
Staphylococcus Pneumococcus
Otitis media
N. catarrhalis
Pneumococcus
Streptococcus l0
Pharyngitis
No growth
No growth
Staphylococcus Pneumococcus
No growth
N. catarrhalis
Staphylcoccus
No growth
Pneumococcus Diphtheroids
N. catarrhalis
Yeast
Staphylcoccus
N. catarrhalis
Pneumococcus 11
Meningitis
i2
Meningitis
13
Otitis media
No growth
N. catarrhalis H. influenzae
Pneumococcus indigo carmine into t h e cerebrospinal fluid and noting the leakage of dye into the right middle ear and out of the nose. On a subsequent admission, Pantopaque was injected into the cisterna magna, and on x-ray examination there appeared to be some dye passing out of the right Eustachian tube. On neurosurgical exploration, closure of a suspected defe~t on the posterior surface of the right temporal bone was attempted with nylon mesh. One month later the child was readmitted with meningitis. Laminagrams in the reverse Towne position showed a linear
fracture line extending into the lateral part of the petrous pyramid. The right mastoid was then re-explored via a radical mastoidectomy. During the operation, dye was injected into the right ventricle and was seen to leak from the roof of the bony part of the right Eustachian tube in the area where the fracture line was visualized on x-ray examination. The bony opening of the right Eustachian tube was then packed with bone wax through the middle ear. Following this procedure there has been no further cerebrospinal fluid
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Number 3
Cerebrospinal fluid otorrhea and recurrent meningitis
3 99
Table I. C o n t ' d Admission
Diagnosis
14
Meningitis
15
Meningitis
16
Gastroenteritis
17
Meningitis
18
Meningitis
Throat culture N. eatarrhalis
Nasopharyngeal culture
Cerebrospinal fluid culture
Staphylococcus Pneumococcus Diphtheroids
Pneumococcus
Pneumococcus Staphylococcus
Pneumococcus
Culture of ear or mastoid
No growth Staphylococcus Streptococcus
Pneumococcus
Coli-aerogenes
Staphylococcus
H. influenzae
H. influenzae
H. influenzae
Pneumococcus 19
Staphylococcus
Meningitis
H. influenzae
H. influenzae
20
Meningitis
Enterococcus
No growth
H, streptococcus
21
Meningitis
22
Meningitis
23
Meningitis
Staphylococcus Diphtheroids
No growth No growth
Staphylococcus
No culture
N. catarrhal~
24
Mastoid revision
Streptococcus
25
Meningitis
Staphylococcus
26
Meningitis
Staphylococcus Enterococcus
27
Meningitis
Staphylococcus Streptococcus
No culture No culture
H. influenzae
H. influenzae
Staphylococcus
N. catarrhalis
28
Craniotomy
29
Meningitis
Pneumococcus Streptococcus Staphylococcus
Pneumococcus Staphylococcus Enterococcns
No growth
No growth
N. catarrhalis
otorrhea, a n d no f u r t h e r meningitis occurred d u r i n g a p e r i o d of 2 years.
has
DISCUSSION T h e most c o m m o n cause of cerebrospinal fluid o t o r r h e a is f r a c t u r e of the skull. 6-9 T h e incidence of cerebrospinal fluid o t o r r h e a in all of these fractures m a y be as high as 20 per cent. y, 10, 11 T h e course of a f r a c t u r e t h r o u g h the t e m p o r a l bone is subject to considerable v a r i a t i o n ; the transverse type of fracture is the most dangerous. This usually involves the b o n y l a b y r i n t h , an a r e a of
ossification in cartilage ( e n d o c h o n d r a l bone) which is, by its nature, predisposed to fibrous union?, 12, is This m a y constitute a persistent r o u t e of infection f r o m the m i d d l e e a r to the cerebrospinal fluid? -a, 14 However, any temp o r a l bone f r a c t u r e m a y result in a fibrous union w i t h d i m p l i n g of t h e d u r a into the f r a c t u r e line which prevents osseous union. T h e r e are certain r a r e instances in which a f r a c t u r e is n o t the cause of r e c u r r i n g meningitis. C a w t h o r n e 5 m e n t i o n e d a child w i t h 18 episodes of otitic meningitis in which the locus minoris resistentiae was a persistent
4 O0
Spitz et al.
p e t r o s q u a m o u s suture. A n o t h e r route of infection m a y be the n a t u r a l a r a c h n o i d a l extensions into the i n t e r n a l a u d i t o r y m e a t u s 2 Evidence of f r a c t u r e is always to be sought, and the f r a c t u r e line m a y be most difficult to detect on x-ray exalnination. 2, 4, 7 T h e r e is no one x - r a y view t h a t will best show fractures of the t e m p o r a l bone. 1~ T h e presence of cerebrospinal fluid otorr h e a m a y n o t be easy to establish. T h e spinal fluid m a y be m a s k e d b y pus or blood. S o d i u m iodide can be injected into the suba r a c h n o i d space, a n d the presence of iodine in the middle ear can be established within one-half h o u r after the injection. A similar p r o c e d u r e is the use of indigo c a r m i n e inj e c t e d into the cerebrospinal fluid. A r a d i o p a q u e substance can also be instilled and the aid of the x - r a y enlisted in tracing the p a t h of otorrhea. I n the 10 instances of meningitis in which an organisms was isolated f r o m the cerebrospinal fluid, a p n e u m o c o c c u s was isolated 6 times, Hemophilus influenzae 3 times, and the meningococcus once. I n 6 instances the organism isolated f r o m the cerebrospinal fluid was also f o u n d in the n a s o p h a r y n x or throat. Since the route of infection was via the E u s t a c h i a n tube, one w o u l d expect a closer correlation between the cultures from the spinal fluid and the nose a n d throat. Several studics have shown t h a t cultures from the u p p e r r e s p i r a t o r y t r a c t have little v a l u e in p r e d i c t i n g the etiological agent of otitis media. 16, 1~ CONCLUSIONS F o r the definitive t h e r a p y of each episode of meningitis, it is most i m p o r t a n t t h a t cultures of the cerebrospinal fluid be obtained. I n this case of cerebrospinal fluid otorrhea, bacteriologic studies of the u p p e r respiratory t r a c t were of little value in delineating the t h e r a p y of a c o m p l i c a t i n g meningitis. L a m i n a g r a p h y is an essential study in searching for a possible f r a c t u r e as the p a t h of cerebrospinal fluid otorrhea. If the site of the leak into the ear is not d e t e r m i n e d p r e o p e r a t i v e l y , a r a d i c a l mas-
September 1961
t o i d e c t o m y is essential to visualize the entire roof of the m i d d l e ear a n d the E u s t a c h i a n tube. D y e should be injected into the cereb r o s p i n a l fluid at the time of operation. SUMMARY
This is the r e p o r t of an u n u s u a l case of a child in w h o m otitic meningitis r e c u r r e d t w e n t y times. F o u r surgical p r o c e d u r e s were r e q u i r e d before the route of infection was finally detected a n d obliterated. Bacteriologic studies of the pathogens in the u p p e r r e s p i r a t o r y t r a c t d i d not necessarily show c o r r e l a t i o n with organisms isolated on the c u l t u r e of the eerebrospinal fluid. L a m i n a g r a p h y is essential in searching for a possible f r a c t u r e of the skull as the p a t h of cerebrospinal fluid o t o r r h e a . If the site of leak into the e a r is not d e t e r m i n e d preoperatively, a r a d i c a l m a s t o i d e c t o m y is essential to visualize the entire m i d d l e ear and the E u s t a c h i a n tube. D y e should be injected into the cerebrospinal fluid at the time of operation. REFERENCES
1. Maxwell, H.: Recurrent Otogenic Meningitis, Laryngoscope 63. 355, 1953. 2. Hara, A. J.: Otitic Meningitis in the Antibiotic Era, Ann. Otol., Rhin. & Laryng. 68: 305, 1959. 3. Precechtel, A.: The Problem of Recurrent Meningitis in ORL, Acta oto-laryng. 45: 427, 1954. 4. Muermann, O.: Acta oto-laryng. 45: 429, 1954. 5. Cawthorne, T. E.: Acta oto-laryng. 45: 429, 1954. 6. Dandy, W. E.: Treatment of Rhinorrhea and Otorrhea, Arch. Surg. 49" 75, 1944. 7. Grove, W. E.: Otoneurology of Head Injuries, in Coates, G. M., Schenck, tI. P., and Miller, H. V.: Otolaryngology, Hagerstown, 1959, W. F. Prior Company, Inc. 8. Skolnick, E. M., and Ferrer, J. L.: Cerebrospinal Fluid Otorrhea, Arch. Oto-laryng. 70' 795, 1959. 9. Grove, W. E.: Otologic Symptoms or Complications of Craniocerebral Injury, Surg. Gynec. & Obst. 74: 581, 1942. 10. Besley, F.: A Contribution to the Study of Skull Fractures, J. A. M. A. 66: 345, 1932. 11. Phelps, C.: An Analytical and Statistical Review of 1000 Cases of Head Injury, Am. Surgeon 449, 1902.
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Cerebrospinal fluid otorrhea and recurrent meningitis
12. Voss, O.: Der Chirurgie der Schgdelbasisfrakturen, Leipzig, 1936, Erhfarhungen. 13. Grove, W. E.: Skull Fractures Involving the Ear, Laryngoscope 49: 673, 1939. 14. Nager, F. R.: Late Meningitis Following Fracture of the Labyrinth, in Fowler's Loose Leaf Medicine of the Ear, New York, 1947, Thos. Nelson & Sons.
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15. Shambaugh, G. E., Jr.: Surgery of the Ear, Philadelphia, 1950, W. 13. Saunders Company. 16. Mortimer, E. A., Jr., and Watterson, R_ L.: A Bacteriologic Investigation of Otitis Media in Infancy, Pediatrics 17: 359, 1956. 17. Jones, M.: Comparison of Bacteria From Ear and Upper Respiratory Tract, Arch. Otolaryng. 72: 329, 1960.