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Cerebrospinal fluid otorrhea presenting in complicated chronic suppurative otitis media.



Abstract

Since the introduction of antibiotics, the simultaneous occurrence of multiple intra- and extracranial extracranial

external to the cranial vault.


extracranial convulsions
when the cause of the convulsions is external to the brain, e.g. hypocalcemic tetanic convulsions.
 complications in chronic suppurative suppurative

pertaining to or emanating from suppuration; pus in e.g. suppurative arthritis, bronchopneumonia.
 otitis media (CSOM) has been uncommon. Such a combination is associated with high mortality and morbidity, especially in the pediatric pediatric /pe·di·at·ric/ (pe?de-at´rik) pertaining to the health of children.

pe·di·at·ric
adj.
Of or relating to pediatrics.
 age group. Cerebrospinal fluid (CSF) otorrhea presenting in CSOM is unusual and rare. To the best of our knowledge, only 19 cases of CSF otorrhea in complicated CSOM have been previously reported in the literature. Identifying the site of leak might require a high index of suspicion index of suspicion Medtalk A phrase broadly used to indicate how seriously a particular disease is being entertained as a diagnosis; as an example, there is a high IOS that rapid and unexplained weight loss in an elderly Pt is due to pancreas CA, and a low IOS that . Management of such a case could be challenging in terms of leak closure, disease clearance, and restoration of function. We report a new case of CSF leak in CSOM, and we describe our line of management. Even though CSOM is an uncommon indication for radical mastoidectomy today, such surgery might be necessary for these cases.

Introduction

Patients with chronic suppurative otitis media (CSOM) seldom present with multiple concomitant fulminant ful·mi·nant
adj.
Occurring suddenly, rapidly, and with great severity or intensity, usually of pain.



ful
 complications. Cerebrospinal fluid (CSF) leak following injury to the temporal bone is common, but CSF leak in CSOM in addition to other complications is rare. To the best of our knowledge, only 19 cases of CSOM with CSF leak have been previously reported in the literature. (1-3) In this article, we report a new case of CSF otorrhea and CSOM in a patient with multiple intra- and extracranial complications.

Case report

A 12-year-old boy presented to us in serious condition with right-sided otorrhea of 11 years' duration. During the preceding 10 days, the discharge had suddenly become watery and profuse. The otorrhea had become so severe that it had soaked the patient's pillow when he slept with his head turned to the right. At the same time, the patient also had drowsiness, headache, fever, disorientation, and giddiness. He had also experienced seizures and vomiting during the preceding 2 days, and he reported swelling behind the right ear and in the right side of the neck of 1 week's duration. He had no history of ear surgery or ear trauma.

On examination, the watery fluid could be seen dripping from the ear. On closer inspection, the ear canal was filled with granulation tissue and discharge, and the tympanic membrane was completely perforated. A diffuse swelling was observed in the right mastoid mastoid /mas·toid/ (mas´toid)
1. breast-shaped.

2. mastoid process.

3. pertaining to the mastoid process.


mas·toid
n.
The mastoid process.
 region, and it extended downward to the neck along the sternocleidomastoid muscle. Kernig's sign was positive. Funduscopic examination revealed features of papilledema.

Laboratory investigations revealed that the patient's blood parameters were within normal limits and that he was not immunocompromised. Findings of glucose and protein on biochemical analysis of the ear fluid were consistent with CSF. A lumbar puncture was performed, and the CSF was sent for analysis; the pathologist reported pyogenic pyogenic /pyo·gen·ic/ (-jen´ik) suppurative.

py·o·gen·ic
adj.
1. Producing pus.

2. Of, relating to, or characterized by pyogenesis.
 meningitis. Pure-tone audiometry revealed a severe mixed hearing loss (figure 1). Computed tomography (CT) of the head demonstrated a dehiscent de·his·cence  
n.
1. Botany The spontaneous opening at maturity of a plant structure, such as a fruit, anther, or sporangium, to release its contents.

2.
 sinus plate, sigmoid sinus thrombosis, a cerebellar abscess, and dilation of the lateral and third ventricles, which suggested obstructive hydrocephalus (figure 2). The diagnosis was atticoantral-type CSOM with mastoid abscess, Bezold's abscess, sigmoid sinus thrombosis, meningitis, cerebellar abscess, otitic hydrocephalus hydrocephalus (hī'drəsĕf`ələs), also known as water on the brain, developmental (congenital) or acquired condition in which there is an abnormal accumulation of body fluids within the skull. , and CSF otorrhea.

[FIGURES 1-2 OMITTED]

With the patient under local anesthesia, the mastoid and Bezold's abscesses were drained, and the pus was sent for culture and sensitivity. Pseudomonas aeruginosa was identified in the pus, and the patient was started on intravenous ceftazidime at 1 gram twice daily. His general condition improved, and after a few days we were able to demonstrate the CSF leak from the right ear with the patient's head bent forward (figure 3).

[FIGURE 3 OMITTED]

The patient was scheduled for temporal craniotomy Craniotomy Definition

Surgical removal of part of the skull to expose the brain.
Purpose

A craniotomy is the most commonly performed surgery for brain tumor removal.
 and mastoid surgery under general anesthesia. In the operating room, the right cerebellar abscess was identified and excised completely by a neurosurgeon. The mastoid antrum was opened via a postauricular approach. Extensive cholesteatoma and granulation tissue were found in the mesotympanum, attic, hypotympanum, and mastoid antrum. All ossicles Ossicles
The three small bones of the middle ear: the malleus (hammer), the incus (anvil) and the stapes (stirrup). These bones help carry sound from the eardrum to the inner ear.

Mentioned in: Otitis Media, Stapedectomy
 of the middle ear, including the stapes stapes /sta·pes/ (sta´pez) [L.] the innermost of the auditory ossicles; it articulates by its head with the incus and its base is inserted into the oval window

sta·pes
n. pl.
 footplate, were found to be destroyed. The sinus plate had been destroyed, but the tegmen tegmen /teg·men/ (teg´men) pl. teg´mina   [L.] a covering structure or roof.

tegmen tym´pani
 plate was intact. The sigmoid sinus was solid and filled with granulation tissue. The retrosigmoid dura was found to be involved and breached. Pulsating clear fluid was seen leaking from the site. Two fistulae were discovered, one in the horizontal semicircular canal The lateral or horizontal canal (external semicircular canal) is the shortest of the three canals.

It measures from 12 to 15 mm., and its arch is directed horizontally backward and lateralward; thus each semicircular canal stands at right angles to the other two.
 and one in the superior semicircular canal The introduction to this article provides insufficient context for those unfamiliar with the subject matter.
Please help [ improve the introduction] to meet Wikipedia's layout standards. You can discuss the issue on the talk page.
, and both were filled with granulation tissue (as a result, labyrinthine fistula was added to the list of complications). The eustachian tube orifice, the oval window, and the round window were also filled with granulation tissue. No herniation herniation /her·ni·a·tion/ (her?ne-a´shun) abnormal protrusion of an organ or other body structure through a defect or natural opening in a covering, membrane, muscle, or bone.  of the brain was observed in any part of the mastoid antrum.

In view of such extensive disease and destruction, we were left with no choice but to perform a radical mastoidectomy. The disease was meticulously cleared from the middle ear and mastoid antrum. After the granulation tissue was cleared from the fistulae, they were sealed with bone wax. The eustachian tube orifice was sealed with temporalis muscle tissue. Curettage curettage /cu·ret·tage/ (ku?re-tahzh´) [Fr.] the cleansing of a diseased surface, as with a curet.

medical curettage
 was performed on the sigmoid sinus until all disease was cleared and the dura exposed. The defect was then closed in three layers with temporalis fascia, temporalis muscle tissue, and homograft homograft: see transplantation, medical.  septal septal /sep·tal/ (sep´tal) pertaining to a septum.

sep·tal
adj.
Of or relating to a septum or septa.
 cartilage. The round window and the oval window were covered with temporalis fascia. A meatoplasty was performed in the cartilaginous cartilaginous /car·ti·lag·i·nous/ (kahr?ti-laj´i-nus) consisting of or of the nature of cartilage.

car·ti·lag·i·nous
adj.
1. Chondral.

2.
 external canal. A tube drain was placed to connect the cavity with the external auditory canal external auditory canal
n.
See ear canal.
.

Postoperatively, the CSF leak stopped and the patient improved markedly. He was discharged after 10 days. At the 6-month follow-up, no evidence of recurrence of disease or CSF leak was seen.

Discussion

Chronic otitis media Chronic otitis media
Inflammation of the middle ear with signs of infection lasting three months or longer.

Mentioned in: Myringotomy and Ear Tubes

chronic otitis media 
 is widespread in India and elsewhere in the developing world. Many patients present quite late in the course of their disease, and therefore complications can be quite serious. Despite advances in healthcare delivery, aggressive atticoantral disease with multiple complications is prevalent in many parts of India, primarily in children. One of the Figure 2. Axial CTs of the head she complications that is and the dilation of the ventricles (C not common is CSF leak. In fact, to the best of our knowledge, only 19 cases have been reported. In 1961, Alberti and Dawes reported a series of 6 cases of CSF otorrhea associated with chronic ear disease. (1) In 1980, Hicks et al reported 11 such cases, 7 of which included intracranial abscesses. (2) Finally, in 1983, Adkins and Osguthorpe described the mini-craniotomy procedure for the management of 2 cases of CSF otorrhea from tegmen defects caused by chronic ear disease. (3) To the best of our knowledge, our case represents the 20th such published report.

Very few cases of dural dural /du·ral/ (dur´'l) pertaining to the dura mater.

dural

pertaining to the dura mater.


dural ossification
see dural ossification.
 exposure with resultant herniation of brain tissue and CSF leak as a result of CSOM, with or without cholesteatoma, have been described in the literature.(1,4,5) In many cases, CSF otorrhea in CSOM has been found to occur in association with intracranial abscesses. (2) Cases of CSF otorrhea giving rise to recurrent meningitis have also been described in the literature. (6)

Spontaneous CSF otorrhea through the oval window as a cause of recurrent meningitis has been reported in the recent past. (7) The hallmark of this pathology was the dilation of the cochlea cochlea (kŏk`lēə): see ear.  and the absence of the modiolus modiolus /mo·di·o·lus/ (mo-di´o-lus) the central pillar or columella of the cochlea.

mo·di·o·lus
n. pl. mo·di·o·li
The central conical bony core of the cochlea of the ear.
 on temporal bone. (7) In the inner ear, high-resolution CT of the 7 the potential sites of CSF leak include the oval window, stapes footplate, eustachian tube, and promontory fistula. (7) Leaks from the round window or through defects in the promontory are uncommon. (7) In our patient, no perilymph perilymph /peri·lymph/ (per´i-limf) the fluid within the space separating the membranous and osseous labyrinths of the ear.

per·i·lymph
n.
 gusher was observed from the windows or from any other site in the middle ear, even after the extensive disease was cleared from the area.

According to the literature, the most likely mechanism of CSF otorrhea in CSOM is either (1) the erosive action of the disease on bone, with involvement of the subarachnoid space, or (2) the dehiscence dehiscence /de·his·cence/ (de-his´ins) a splitting open.

wound dehiscence  separation of the layers of a surgical wound.


de·his·cence
n.
 that occurs as a result of previous surgery for preexisting pre·ex·ist or pre-ex·ist  
v. pre·ex·ist·ed, pre·ex·ist·ing, pre·ex·ists

v.tr.
To exist before (something); precede: Dinosaurs preexisted humans.

v.intr.
 chronic ear disease. (8,9) CSF leak is further facilitated by the congenital dehiscence of tegmen, which has an incidence of 5.4%. (10)

The CSF leak in our patient could be attributed only to the CSOM because he had no history of ear surgery or ear trauma. Also, we did not find any brain tissue hemiation along with the CSF leak. Other than the erosive action of the local disease, the simultaneous occurrence of meningitis, cerebellar abscess, and hydrocephalus might have led to an increase in intracranial pressure and facilitated a breach of the inflamed meninges meninges (mĭnĭn`jēz), three membranous layers of connective tissue that envelop the brain and spinal cord (see nervous system). The outermost layer, or dura mater, is extremely tough and is fused with the membranous lining of the skull. .

The pathologic prerequisites for CSF leak in patients with these intracranial complications need to be investigated further. It has been shown that disease in the area of the lateral sinus can destroy the bone of the sinodural angle. (1) Further erosion of the bone over Trautmann's triangle can lead to profuse CSF otorrhea from the lateral cistern. (9) CSF leak might also be made possible by the erosion of the bony plates covering the dura and the subsequent breach of dura through the track of intracranial abscesses. In our patient, no obvious pathway into the intracranial abscess existed, either from the mastoid antrum or from the middle ear. The diagnosis of CSF otorrhea with CSOM is not difficult when it is suspected. The presence of clear fluid in a chronically draining ear that has been cleaned under the microscope is almost diagnostic. (2)

The definitive surgical procedure for these cases is often dictated by the specific individual conditions at the time of surgery. (2) Mastoid surgery is usually the initial procedure, along with drainage of the abscesses. Radical mastoidectomy was performed in 3 of the 11 cases of CSF otorrhea with chronic ear disease and temporal lobe abscess reported by Hicks et al. (2)

In the absence of brain tissue hemiation, surgical repair of the leak site in the dura can be performed conveniently via a transmastoid approach. Repair is preferably done in layers after all disease has been cleared. If the defect is small, temporalis fascia will suffice, but larger defects require fascia lata reinforced with temporalis muscle and cartilage to hold the fascia in place. Certain synthetic biomaterials, such as Marlex mesh, have also been used to support the fascia in cases of brain herniation. (2) It is difficult--and unnecessary--to suture these layers to the dura in a transmastoid repair, but care should be taken to tuck the fascia deep to the edges of the bony defect and then to pack it well into the space. (2) A craniotomy approach has been described for middle fossa fossa /fos·sa/ (fos´ah) pl. fos´sae   [L.] a trench or channel; in anatomy, a hollow or depressed area.

acetabular fossa  a nonarticular area in the floor of the acetabulum.
 defects, especially in cases of CSF otorrhea with brain herniation. (1)

In conclusion, a number of cases of spontaneous, congenital, and traumatic CSF otorrhea have been reported in the literature, but few have been reported with complicated CSOM. The possibility of CSF otorrhea must be kept in mind in a case of CSOM when the ear discharge suddenly turns watery, especially in the presence of intracranial complications. If the diagnosis of CSF otorrhea is missed and thus not adequately addressed by surgery, both recurrent CSF leaks and meningitis could ensue. Our case was unique in that it was managed successfully despite the fact that the patient presented at an advanced stage with multiple complications. CSF otorrhea in CSOM is one of the few non-neoplastic indications for a radical mastoidectomy today.

Acknowledgments

The authors thank Dr. Khaja Naseeruddin, professor and head of the Department of Otolaryngology-Head and Neck Surgery at the Kamataka Institute of Medical Sciences in Hubli, India, for his support and permission to submit this paper for publication.

References

(1.) Alberti PW, Dawes JD. Cerebrospinal otorrhoea in chronic ear disease. J Laryngol Otol 1961;75:123-35.

(2.) Hicks GW, Wright JW Jr., Wright JW III. Cerebrospinal fluid otorrhea. Laryngoscope 1980;90(suppl 25): 1-25.

(3.) Adldns WY, Osguthorpe JD. Mini-craniotomy for management of CSF otorrhea from tegmen defects. Laryngoscope 1983;93:1038-40.

(4.) Dedo GG, Sooy FA. Endaural encephalocele and cerebrospinal fluid otorrhea. Ann Otol Rhinol Laryngol 1970;79:168-77.

(5.) Lundy LB, Graham MD, Kartush JM, LaRouere MJ. Temporal bone encephalocele and cerebrospinal fluid leaks. Am J Otol 1996;17:461-9.

(6.) Harris HH. Cerebrospinal otorrhea and recurring meningitis: Report of three cases. Laryngoscope 1978;88:1577-85.

(7.) Teo DT, Tan TY, Eng SP, Chan YM. Spontaneous cerebrospinal fluid otorrhoea via oval window: An obscure cause of recurrent meningitis. J Laryngol Otol 2004;118:717-20.

(8.) Cawthorne T. The surgery of the ear in the management of intracranial complications of chronic suppurative otitis media. J Laryngol Otol 1955;69:579-87.

(9.) Blatt IM. Surgical repair for cerebrospinal otorrhea due to middle ear and mastoid disease. Report of six cases. Laryngoscope 1963;73: 446-60.

(10.) Balance CA, Green CD, eds. Essays on the Surgery of the Temporal Bone. Vol. I. Plate XIII, figure 1. London: Macmillan & Co.; 1919.

Vikram Bhat, MS, DNB, MNAMS (ORL-HNS); Dandinarasaiah Manjunath, MBBS

From the Department of Otolaryngology-Head and Neck Surgery, Karnataka Institute of Medical Sciences The Karnataka Institute of Medical Sciences (KIMS) is a medical college in Hubli, Karnataka, run by the Government of karnataka state. The institute was established in 1957. Prior to 1996, the institute was known as the Karnataka Medical College (KMC). , Hubli, Karnataka, India.

Reprint requests: Dr. Vikram Bhat, No: 4-64, Sagri Nole, Kunjibettu Post, Udupi-576102, Karnataka, India. Phone: 91-836-237-0579; cell: 91-944-865-8213; e-mail: entvikram@rediffmail.com
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Author:Manjunath, Dandinarasaiah
Publication:Ear, Nose and Throat Journal
Date:Apr 1, 2007
Words:2176
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