Case study spontaneous otoliquorrhea in adult

Abstract

Introduction: The spontaneous otological CSF leaks are rare entities. Usually they manifest themselves in children with meningitis and deep sensorineural hearing loss and in adults with middle otitis and effusion clinical picture. Objective: To describe the clinical picture, diagnosis and surgical treatment of a patient with CSF otological. Case Report: A female sex patient, 57 years old, with hearing loss, aural fullness and right ear tinnitus case history 10 years ago. After insertion of tympanostomy tubes on the right in another service, it began liquid, transparent and constant otorrhea in large quantities. The diagnosis was performed by suggestive cisternoscintigraphy of CSF leak, and by cisternography that showed that the area of bone dehiscence in the tegmen tympani region to the right, partial opacification of mastoid cells and middle ear to the right, besides concentration of contrast medium in sub-arachnoid space to the lower right compared to the left side. Through transmastoid via was located the CSF leak and meningoencephalocele in the region of the right tegmen tympani. The closure of the fistula was accomplished through the use of the temporal muscle flap, fibrin glue and Surgicel®. Final Comments: In adults with symptoms suggestive of recurrent otitis media with effusion, the diagnosis of CSF fistula should be raised. The research should proceed with imaging, highlighting the CTC. Surgical treatment in this patient using the transmastoid technique has proved effective in the short and long term.

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Case Study Spontaneous Otoliquorrhea in Adult
Relato de Caso: Otoliquorreia Espontânea em Adulto
Amanda Costa Rossi*, Juliana Martins de Araujo Cardoso Bertoncello**, Luiz Carlos Scachetti***,
Carolina Schäffer***, Aguinaldo Pereira Catanoce****, Silvio Antonio Monteiro Marone*****.
* Resident of the Year 3 of the Department of Otolaryngology, Hospital Celso Pierro School of Medicine, PUC Campinas.
** Otorhinolaryngologist. Specialist Medical Assistant Department of Otolaryngology, Hospital Celso Pierro School of Medicine, PUC Campinas.
*** Medical (a) ENT. Medical (a) Specialist Contributor (a) of the Department of Otolaryngology, Hospital Celso Pierro School of Medicine, PUC Campinas.
**** MD Neurosurgeon. Specialist Assistant to the Department of Neurosurgery, Hospital Celso Pierro School of Medicine, PUC Campinas, Vera Cruz Hospital and the
Hospital Estadual Mário Covas FMABC.
***** Professor of Otolaryngology, School of Medicine, PUC Campinas. Professor of Otolaryngology, School of Medicine, PUC Campinas and Professor of Otolaryngology,
School of Medicine, USP.
Institution:Celso Pierro Hospital and Medical School, PUC Campinas.
Campinas / SP - Brazil.
Mail adress: Amanda Rossi Costa - Rua José Luis Pereira de Com Queiros, 170 - Apto 141 - Botafogo - Campinas / SP - Brazil - Zip code: 13020-080 - Telephone:
(+55 19) 3343-8599 - E-mail: amandarossi@hotmail.com
Article received on May 3, 2009. Article accepted on August 4, 2009.
SUMMARY
Introduction:
The spontaneous otological CSF leaks are rare entities. Usually they manifest themselves in children
with meningitis and deep sensorineural hearing loss and in adults with middle otitis and effusion
clinical picture.
To describe the clinical picture, diagnosis and surgical treatment of a patient with CSF otological.
A female sex patient, 57 years old, with hearing loss, aural fullness and right ear tinnitus case history
10 years ago. After insertion of tympanostomy tubes on the right in another service, it began liquid,
transparent and constant otorrhea in large quantities. The diagnosis was performed by suggestive
cisternoscintigraphy of CSF leak, and by cisternography that showed that the area of bone dehiscence
in the tegmen tympani region to the right, partial opacification of mastoid cells and middle ear to the
right, besides concentration of contrast medium in sub-arachnoid space to the lower right compared
to the left side. Through transmastoid via was located the CSF leak and meningoencephalocele in the
region of the right tegmen tympani. The closure of the fistula was accomplished through the use of
the temporal muscle flap, fibrin glue and Surgicel®.
In adults with symptoms suggestive of recurrent otitis media with effusion, the diagnosis of CSF fistula
should be raised. The research should proceed with imaging, highlighting the CTC. Surgical treatment
in this patient using the transmastoid technique has proved effective in the short and long term.
CSF otorrhea, fistula, meningitis.
Objective:
Case Report:
Final Comments:
Keywords:
RESUMO
Introdução:
As fístulas liquóricas otológicas espontâneas (FLOEs) são entidades raras. Geralmente manifestam-se
em crianças com meningite e perda sensorioneural profunda e em adultos com quadro de otite média
com efusão.
Descrever o quadro clínico, o diagnóstico e o tratamento cirúrgico de uma paciente com FLOE.
Paciente do sexo feminino, 57 anos, com história de hipoacusia, plenitude aural e tinnitus à direita
há 10 anos. Após colocação de tubo de ventilação à direita em outro serviço, iniciou otorreia líquida,
transparente e constante em grande quantidade. O diagnóstico foi realizado através da
cisternocintilografia, sugestiva de fístula liquórica; e da cisternotomografia que mostrou área de deiscência
óssea em região de tegmen tympani à direita, velamento parcial de células mastóideas e de orelha
média à direita, além concentração do meio de contraste no espaço sub-aracnoídeo à direita menor
em relação ao lado esquerdo. Através de via transmastoídea foi localizada fístula liquórica e
meningoencefalocele na região de tegmen tympani à direita. O fechamento da fístula foi realizado
através do uso de retalho de músculo temporal, cola de fibrina e Surgicel®.
Em adultos com história clínica sugestiva de otite média com efusão recorrente, a hipótese diagnóstica
de fístula liquórica deve ser levantada. A investigação deve prosseguir com exames de imagem,
destacando-se a cisternotomografia. O tratamento cirúrgico nesta paciente, através da técnica
transmastoídea, se revelou eficaz a curto e a longo prazo.
otorreia de líquido cefalorraquidiano, fistula, meningite.
Objetivo:
Relato do Caso:
Comentários Finais:
Palavras-chave:
Case Report
Intl. Arch. Otorhinolaryngol., São Paulo - Brazil, v.14, n.4, p. 472-476, Oct/Nov/December - 2010.

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Case study spontaneous otoliquorrhea in adult.Rossi et al.
Intl. Arch. Otorhinolaryngol., São Paulo - Brazil, v.14, n.4, p. 472-476, Oct/Nov/December - 2010.
INTRODUCTION
The otologic cerebrospinal fluid leakage was mainly
due to acquired factors such as temporal bone fractures
and surgeries involving the temporal bone (1).
Otological spontaneous CSF fistulas (floe) are rare
and can be grouped according to age into two groups
(1,2,3):
• In children due to the presence of failures in the
temporal bone as dehiscence. In these cases an early
manifestation is between one and five years of life,
and usually presents with meningitis and profound
sensorineural hearing loss.
• In adults in the fifth decade of life, generally obese
women with clinical secretory otitis media. It presents
one or more areas of bony dehiscence in the
tegmen tympani and less commonly in the posteri-
or fossa.
The pathophysiology of the floe is not yet fully
understood.
There are two theories responsible for the congenital
and the presence of arachnoid granulations:
• The congenital bone defect theory posits that small
communications with the tegmen result from
anomalous embryological development (4). The
passage of CSF may occur due to inner ear to the
cochlear aqueduct and thus extended through a defect
in the modiolus which allows passage through the oval
or round windows (5), or directly to middle ear
through changes in the cleft of timpanomeníngea
HYRTL (6).
• The presence of granulation theory was postulated by
RICHARD GACEK in 1990 (7) and suggests that arachnoid
granulations during embryonic development ending
in a blind bottom surface inner bone of the skull base.
The central portion of these granules contains liquor
and covered loosely by a fibrous capsule and the
endothelium. Over time, pressure from the
subarachnoid space causes erosion of the fibrous
capsule of the nasal bone and consequently the
anterior, middle or later, resulting in passage of CSF to
the middle ear.
The diagnosis of floe depends largely on clinical
suspicion. It is suggested that any patient over 50 years
with symptoms suggestive recurrent otitis media with
effusion should be taken as a diagnosis among other floe
(4).
The aim of this paper and describe the clinical
features, diagnosis and surgical treatment of a patient into
a floe Otorhinolaryngology Service of a tertiary care
hospital, with residency in Otolaryngology.
CASE REPORT
Female patient, 57 years, sought the Department
of Otolaryngology with a history of hearing loss, ear
fullness and tinnitus on the right for about 10 years. He
denied vertigo. Nearly a year had been submitted to the
use of a ventilation tube to the right in another service.
Since then started otorrhea liquid, transparent and
consistent in great quantity, without improvement with
the use of eardrops. Personal history included arterial
hypertension, diabetes mellitus and asthma. He denied
any history of meningitis, trauma or other otologic surgery.
ENT examination was normal except for right
otoscopy, which showed opaque tympanic membrane
with the presence of a ventilation tube through which
drained large amounts of clear liquid.
The endoscopy showed no abnormalities, tubal
ostia bilaterally free.
She underwent the following complementary
exams.
The dosage of glucose ear fluid was 1 mg / dL
(from the ventilation tube was still inserted), which
suggested it is not liquor.
Audiometry, had mild conductive hearing loss on
the right.
As for imaging, computed tomography of the
temporal bone showed no bone dehiscence. Magnetic
resonance imaging did not provide relevant data.
It was then submitted to cistern cintilography with
signs suggestive of CSF, showing concentration of
radiotracer in the region of the right ear (Figures 1, 2 and
3). CTC, after the introduction of intrathecal iodinated
contrast medium showed the area of bony dehiscence in
the region of the right tegmen, partial opacification of the
mastoid cells and middle ear to the right (Figure 4).Also
showed asymmetric concentration of contrast medium in
the subarachnoid space, showing up in smaller quantities
in the right cerebral hemisphere (Figures 5 and 6), thus
confirming the hypothesis of CSF fistula.
Through transmastoid CSF leak was located about
1 mm in the right tegmen tympani, which is also
evidenced meningoencephalocele the same region.
The closure of the fistula was achieved through the
placement of a temporalis muscle flap, followed by
placement of fibrin glue and Surgicel®. After this
procedure, was removed the vent tube right tympanic

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Figure 1. Cisternocintilografia - Previous - Suggestive of
cerebrospinal fluid leak in the area of the right ear (arrow).
Figure 2. Cisternocintilografia - Rear - Suggestive of
cerebrospinal fluid leak in the area of the right ear (arrow).
Figure 3. Cisternocintilografia - Right Side - Suggestive of
cerebrospinal fluid leak in the area of the right ear (arrows).
Figure 4. Cisternography of the temporal bone (coronal) -
area of bony dehiscence in the tegmen tympani (arrow).
membrane. We opted for the withdrawal of ventilation
tubes for while maintaining the same power to highlight
the possible permanence of otorrhea, this could be
ascertained by otoscopy by changes of the tympanic
membrane . Moreover, the permanence of the vent
pipe could facilitate the onset of infection.
The patient remained 72 hours with lumbar drainage
in the supine position with head elevated on bedrest in
diet and laxative. During his admission he used an IV
Ceftriaxone 2g of 12/12 hours. The patient was discharged
on the fifth postoperative day. In the immediate
postoperative otorrhea had no more liquid and remained
Case study spontaneous otoliquorrhea in adult.Rossi et al.
Intl. Arch. Otorhinolaryngol., São Paulo - Brazil, v.14, n.4, p. 472-476, Oct/Nov/December - 2010.

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so during follow-up that continues to be conducted
periodically for 12 months.
Postoperative audiometry performed, which
revealed no abnormalities.
DISCUSSION
Age and clinical presentation of this case, are
consistent with the theory of arachnoid granulation due to
the age of onset and type of clinical presentation. Regarding
gender, most studies (2,3,8) show a higher incidence in
middle-aged women. The susceptibility in women is not
well understood, increased intracranial pressure is suggested
as a possible cause in these women (2). There is still no
published data that relate to the female floe (9).
Early diagnosis of floe depends on a high degree of
clinical suspicion (2,3,4,9). In our case, the suspect took
place after the completion of the clinical exam, with no
explanation for the occurrence of such otorrhea.
Can be performed laboratory tests such as fasting
glucose level of the liquid. A concentration of 30 mg / dL
glucose is considered positive CSF in a patient with normal
glucose level. However, the chemical analysis of fluid
obtained from the ear depends on the presence of
contamination or infection, bringing a high rate of false
positive. In our case, the fasting glucose level was 0.1 mg
/ dL, it is not conclusive for diagnosis. highly specific and
sensitive to CSF leak is the dosage of beta 2 transferrin, a
specific protein from the CSF and can be measured in
extremely small amounts of fluid (4), being an expensive
test. We do not have in our service.
In evaluating patients with suspected CSF leak at
high resolution CT of the temporal bone is considered
essential. MRI reveals no bone dehiscence. The intrathecal
injection of radioisotope was helpful in diagnosing the
presence of fistula, but no help in locating the site (9).
Regarding surgical approach, several authors refer
to the technique of the middle fossa craniotomy for repair
of fistula. There are also recent reports of access transmastoid
and combination of these approaches (1,2,3,9,10).
The middle fossa approach provides wide exposure,
so dehiscence (larger than 2 cm), multiple or extending
anteriorly to the petrous apex can be corrected by this
route (4), saving your hearing. However, it is a procedure
with great potential for complications (10). The
transmastoid has the advantage of accessing the middle
and posterior fossa craniotomy and preventing retraction
of the temporal lobe (4). It is also technically easier to
perform, and a procedure with fewer risks and
Figure 5. Cisternography temporal bone (axial) - The
concentration of contrast medium to a lesser extent in sub-
arachnoid space to the right over the left side (arrow).
Figure 6. Cisternography temporal bone (axial) - The
concentration of contrast medium to a lesser extent in sub-
arachnoid space to the right over the left side (arrow).
Case study spontaneous otoliquorrhea in adult.Rossi et al.
Intl. Arch. Otorhinolaryngol., São Paulo - Brazil, v.14, n.4, p. 472-476, Oct/Nov/December - 2010.

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complications, not proving a deterrent for the correction
of bone defects, even larger. Thus, you can choose
initially by transmastoid and failure of this procedure
using a broader access to the craniotomy (10). In our case,
we chose to perform the transmastoid as the first option
to be a less invasive procedure.
Several materials can be used for repair of CSF
leaks: as autologous fascia, muscle, cartilage, fat and bone,
and various combinations of materials and synthetic
counterparts (4, 10). The closure technique with multiple
layers has shown higher rates of permanent closure and
lower recurrence rate (2.4). In our case we used the
technique of closure with multiple layers, with the
temporalis muscle, fibrin glue and Surgicel®.
A lumbar shunt can be used as an adjunct for
postoperative decrease the intracranial pressure. Its use
in the postoperative period is controversial due to risk of
potential complications such as meningitis, headache or
catheter obstruction (10). In the case described lumbar
drainage was used for 72 hours without complicações.O
use of antibiotic prophylaxis is not mentioned by many
authors, but there are reports on the use of Ceftriaxone 1g
8 / 8 hours (10). In our case we chose Ceftriaxone 2g 12/
12 hours as a prophylactic measure.
CONCLUSION
Otologic cerebrospinal fluid leak may occur in
the absence of a history of trauma or meningitis. In
adults with symptoms suggestive of recurrent otitis
media with effusion, especially with no cause for such,
the diagnosis of CSF should be raised. The investigation
should proceed with imaging, highlighting the CTC.
Surgical treatment in this patient, using the technique
transmatoid proved effective in the short and long
term.
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Cerebrospinal Fluid Otorrhea: Diagnosis and Management.
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BW, Rosenberg SI. Diagnosis and Management of
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Case study spontaneous otoliquorrhea in adult.Rossi et al.
Intl. Arch. Otorhinolaryngol., São Paulo - Brazil, v.14, n.4, p. 472-476, Oct/Nov/December - 2010.