Otogenic temporal lobe brain abscess with Gradenigo syndrome: a case report

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Department of ENT Diseases, Yerevan State Medical University after M. Heratsi

Department of Radiology

Department of Neuorosurgery, Astghik MC

Ultra-imaging Scientific Methodological Center of Radiology

Otogenic temporal lobe brain abscess with Gradenigo syndrome: a case report

Nahapetyan N.R., Kirakosyan A.A., Mesropyan H.B., Hambardzumyan A. G., Badjul M.1., Azaryan T. V., Kocharyan A. G.

Intracranial complications as a result of otogenic infections occur even in the antibiotic era. Meningitis is the most common reported intracranial complication, followed by brain abscess and lateral sinus thrombosis. Otogenic intracranial complications may be secondary to acute or chronic otitis media (OM) with or without cholesteatoma. Two or more complications may coexist; hence, evidence of any complications requires ruling out others. We are reporting an unusual simultaneous complications of chronic otitis media-temporal lobe abscess accompanied with Gradenigo syndrome, caused by Granulicatella adiacens in a 26-year old female.

Case report: 26-year old female applied to ENT department of Astghik Medical Center in March, 2018 with complaints of purulent discharge from both ears, convergent strabismus, diplopia, headache, severe pain behind right eye. Patient had history of bilateral chronic otitis media in more than 5 years of duration. A viral infection of the upper respiratory tract was registered one week ago, before admission to the hospital. There was no history of seizures, trauma or rash.

Presentation of the main material

On examination, the patient was conscious and well oriented. Otomicroscopically, there were purulent discharge in her both ears, with central tympanic membrane perforations. After the suction of discharge, the right tympanic cavity was filled of pale granulation tissue, which was visualized especially on the promonto- rial wall, Eustachian tube orifice, and hypotympanum, and grasps the auditory ossicles as well. Neurologic examination revealed neck rigidity, pathological reflex of Babinsky on the left side, focal neurological deficit of granulations, covered the ossicular chain, oval and round windows and the Eustachian tube orifice.

After the suction the edge of perforation was granulous and thickened. Middle ear cavity was filled with unusual pale needle and removed. Mastoidectomy had been performed. Mastoid process was sclerotic. Antrum was filled with purulent discharge and pale granulation tissue, extending till the epitympanum. Head of the malleus and body of incus were covered with granulation as well. Separation of incudostapedial joint was performed, the malleus was cut and both head of the malleus and incus were removed. All anterior epitympa- num was filled with pus and pale granulation, which was removed. Tegmen of anterior epitympanum was rough, but intact. Posteriosuperior bony wall of external ear canal was removed till the facial ridge. A small area of tegmen antri corresponding to the area of brain abscess, that was thin and eroded, was opened. Dura was intact. Mastoid cavity was flattened with diamond burr. The lower part of mastoid cavity was filled with bone pate and covered with fascia.

The next day patient mentioned diminishing of headache and pain behind right eye, but in several day pain was increasing. Bacteriological examination revealed of presence of Granulicatella adiacens in aspirated pus, which was susceptible to amoxiclav, mox- icin, gentamicin, ciprofloxacin, tetracycline, vancomycin and linezolid. Although i/v antibiotical therapy had been done during two weeks after the aspiration of abscess, control imaging revealed accumulation of pus in abscess cavity within the right temporal lobe. On 16^th day after primary surgery. total removal of abscess had been done. A 6x5cm craniotomy of right temporal bone was performed and a bone flap was removed. There was no cerebral pulsation. After opening the dura matter was seen the cerebral edema. The cortex of inferior temporal gyrus was coagulated and cut. In depth of 8mm the solid, adhered wall of abscess capsule was visualized. After freeing the capsule from all sides, the total removal was performed without any damage to capsule wall (fig. 8, 9). meningitis brain abscess otogenic

VI cranial nerve (convergent strabismus, diplopia). There was no papilloedema on fundoscopy. Her speech was normal. Her abdominal and cardiorespiratory examinations were unremarkable.

Her routine laboratory investigations were all within normal limits. The patient was not seropositive for the HIV antibodies. The contrast enhanced CT scan revealed a hypodense lesion with oval enhancement in the right temporoparietal region with surrounding oedema.

CT scan and MRI had been performed (fig. 1-7). Presence of soft tissue within the middle ear cavity revealed, involving auditory ossicles. Ossicular chain was not interrupted. Tympanic cavity mucosa was thickened. There was a zone of homogenious edema of the temporal lobe brain tissue. On the T2 and T1 coronal MRI images, in the right temporal lobe there was irregularly shaped cavity, with heterogeneous content and edema of surrounding brain tissue. In post contrast images, in the right temporal lobe the walls of the cavity where enhanced, inferior wall of the cavity had been closely adjacent to the surface of the temporal bone, in the zone, which corresponds with the bone erosion sights, found on CT images. On the coronal images there was an asymmetrical thickening of dura in the apex of right pyramid. Diagnosis was chronic otitis media, complicated with temporal lobe brain abscess and petrositis /Gradenigo syndrome/.

At the same day patient underwent surgery. After the right temporal bone craniotomy, the abscess cavity was verificated under the ultrasonic control. The punction of cavity was performed by cerebral cannula. 10- 12cc viscous content was aspirated. The cavity was irrigated with saline solution. After that radical mastoidectomy had been performed.

Surgical findings were as follows: there were subtotal perforations of tympanic membrane with purulent discharge These granulations were incised with Rosen Patient was extubated, no neurological deficitis took place. During two postopoperative weeks patient had been done intravenous and 3 months peros antibiotical therapy/ciprofloxacin/

Figure 1. Primary CT scan March 21, 2018

On the primary CT scan there is a soft tissue density in the middle ear cavity, involving ossicles (yellow arrow in transverse plane). Ossicle chain is not interrupted, no signs of destruction. There are no signs of bulging tympanic membrane or tympanic perforation, but tympanic membrane is thickened (yellow arrow in coronal plane).

Figure 2. Primary CT scan March 21, 2018

There are signs of mastoid air cell trabecular sclerosis (red arrow) and partial erosion of the external lamina of the mastoid process in its anterior-superior surface (yellow arrow).

Figure 3. Primary CT scan March 21, 2018

Figure 4. Primary brain contrast enhanced MRI scan March 21, 2018

On the soft tissue window images there is a zone of in homogenious edema of the temporal lobe brain tissue. Patient had neurological complaints, brain MRI was performed.

On the MR T2 tr and T1 coronal images, in the right temporal lobe there is an irregularly shaped cavity, with heterogeneous content and edema of surrounding brain tissue.

Figure 5. Primary brain contrast enhanced MRI scan March 21, 2018

On the post contrast images, in the right temporal lobe the walls of the cavity are enhanced, inferior wall of the cavity is closely adjacent to the surface of the temporal bone, in the zone, which corresponds with the bone erosion sights, found on CT images (yellow arrows on coronal and sagittal plane images).

Figure 6. Primary brain contrast enhanced MRI scan March 21, 2018

On the coronal postenhancenced images there is an asymmetrical thickening of the dura matter.

Figure 7. Primary brain contrast enhanced MRI scan March 21, 2018

On the coronal postenhancenced images there is an asymmetrical thickening of the dura matter.

Figure 8. Intraoperative view After opening the dura mater was seen the cerebral edema.

Figure 9. Removed abscess from the temporal lobe

a b c

Figure 10(a,b,c). Postoperative cystic changes in the right temporal lobe with signs of regression.

Petrous apex without signs of destruction

Discussion

Complications of acute, or chronic, suppurative ear disease manifest acutely and are medical or surgical emergencies because of impending, or concurrent, morbidity or mortality. These complications are defined as a spread of infection beyond the confines of the pneumatized spaces of the temporal bone and the attendant mucosa. Complications are classified into two groups: aural (intratemporal) and intracranial. Aural complications include (1) mastoiditis, (2) petrositis, (3) labyrinthitis, and (4) facial paralysis. Intracranial complications include (1) extradural abscess or granulation tissue, (2) dural venous sinus thrombophlebitis, (3) brain abscess, (4) otitic hydrocephalus, (5) subdural abscess, and (6) meningitis [1]

The majority of intracranial complications were caused by chronic otitis media and cholesteatoma (95.8%), and these complications occur more frequently in the first three decades of life with a higher incidence in males [2]. The commonest to occur are meningitis and brain abscess (temporal or cerebellar) and one or more complications may present in a single patient [3, 4]. They may present with headache, neck stiffness, vomiting, which are caused by the increased intracranial pressure. Seizures have been reported in up to 50% of the cases and fits associated with otorrhea and decreased hearing. However, these may be difficult to recognize and present atypically and more subtly as the symptoms can be masked by use of antibiotics.

Focal neurological deficits which are related to the site of the abscess may be present, depending on the site of the lesion. Gradenogo's Syndrom is an uncommon but life-threatening complication of otitis media. The typical presentation of GS comprises a sixth cranial nerve palsy, otorrhea, headache, and pain along the distribution of the trigeminal nerve. Most cases of petrous apicitis do not present with the classic clinical triad however [5, 6]. The time interval between the onset of otitis media and the clinical presentation of abducens nerve palsy varies from 1 week to 2 to 3 months [7].

The most powerful, rapid, efficient, and useful diagnostic tools are the expertly performed history and physical examination. Complete diagnoses are made from the history, the physical examination, and surgical exploration. Careful surgical observations, through thin bone, of the dura at the tegmen, the sigmoid sinus, and the facial nerve, are often crucial for complete diagnosis. Imaging is important to observe bone changes using computed tomography (CT) and soft tissue changes using magnetic resonance imaging (MRI). However, reliance on imaging modalits alone, at the expense of the above-mentioned three elements, carefully done, may lead to serious errors. For example, papilledema is easily missed if fundoscopic examination is not done or large epidural abscesses may be missed if intraoperative dissection does not include looking for diseased dura, through thin bone, over the tegmen sigmoid, and posterior fossa plate.

Petrositis often this will resolve with systemic antibiotics and a modified radical or radical mastoidectomy, removing as many air cells toward the petrous apex as possible. Occasionally, an extensive three-dimensional approach, including a middle fossa approach to the petrous apex, is required [10].

Our case presented with complaints of headache and a past history of chronic bilateral ear discharge. There were signs of a focal neurological deficit (convergent strabismus, diplopia). Brain scans, CT and MRI are the important tools that enable in making an accurate diagnosis of the infection. In this patient, the diagnosis of the brain abscess was established by CT scan and MRI. The definitive microbiological diagnosis was made by the submission of the pus from the abscess. Aspiration of the pus provides the best opportunity to make a microbiological diagnosis and also to report an optimal therapy. The bacteriology of an otogenic abscess represents the microbial flora of a chronically infected ear. The isolates from the abscess include Streptococci (aerobic and anaerobic like Peptostreptococcus), Bacteriodes fragilis, Enterobacterieaceae species esp. Proteus species and Actinomyces species. In our patient, the aetiological agent was Granulicatella adiacens which rarely causes otogenic brain abscess.

Granulicatella adiacens is a Gram-positive coccus, formerly grouped with nutritionally variant Streptococcus, often found as commensal bacteria of the human oral cavity, urogenital tract, and gastrointestinal tract. Granulicatella spp. as a pathogen can cause bacteremia and infective endocarditis particularly of prosthetic valves and pacemaker leads. Bacteremia with Granu- licatella spp. without endocarditis has been reported, including a single case of infection of aortic atheroma with associated dissection [8]. Other infections have included seeding by prosthetic material or surgery with isolates from brain abscess, CSF, joint space, vertebrae, and breast implant [8].

Thus, Granulicatella spp. is an uncommon cause of otogenic brain abscess and its appropriate identification is required to diagnose the case, so that an early treatment can be given to prevent complications. Antibiotics are very effective in the early and later stages of cerebritis [9]. In our patient, Granulicatella spp. was susceptible to amoxiclav, moxicin, gentamicin, ciprofloxacin, tetracycline, vancomycin and linezolid. The ineffectiveness of antibiotics in the stage of the capsule formation is due to the acidic medium within the abscess cavity and the inability to have adequate therapeutic concentration of the antibiotic within the abscess. Therefore, a surgical intervention is essential once the capsule is well formed [9]. For our patient, burr hole tapping was done with tympanomastoidec- tomy and broad spectrum intravenous antibiotic therapy, but she didn't respond markedly. Later, temporal craniotomy and abscessectomy was done (Fig. 8,9). Right temporal lobectomy is a safe procedure. The right side lobe is usually non-dominant. It can be safely resected upto 6 cm from temporal apex. You will not get any deficit. But resection more than this can cause memory impairment and impairment of extra-curricular activities.

Our patient is doing well as was observed on his follow up visits, with no residual neurological deficit, with a marked improvement. Postoperative CT scan (after 4 weeks) shows regression of the lesion with no midline shift, petrous apex without signs of destruction. (fig. 10a, b, c,).

Conclussion

The most powerful tools of diagnosis are an expertly performed, detailed, and time-specific history and physical examination. Imaging is the only way to detect brain abscess and subdural abscess. A complete diagnosis may ultimately depend on carefully done intraoperative observations. Complications of suppurative ear disease manifest in obvious or predictable patterns that help guide preoperative diagnosis and intraoperative discovery and treatment. Treatment of complications requires hospitalization, culture-specific intravenous antibiotics, expedient surgical exenteration of the ear disease, and a specific tailored approach to the complication.

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