Case study
Late complication of cochlear implantation: Necrotizing meningoencephalitis
Complicación tardía de implante coclear: meningoencefalitis necrosante
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She was implanted at the age of 9 years, using a classic facial recess approach, with no intraoperative or immediate postoperative complications. She responded well to the implant, with very good audiometric results and open discrimination in free field.</p><p id="par0010" class="elsevierStylePara elsevierViewall">In November 2019, she presented urgently to paediatrics with facial paralysis (grade IV House-Brackmann) and intense intermittent holocranial headache. She underwent a multislice computed tomography (CT) scan of the brain which was reported as normal; she was therefore prescribed meprednisone and vitamin B12 for facial palsy. The patient was treated and managed by the paediatrics service, and the general medical history and otoscopic examination revealed the presence of an intact, congestive eardrum.</p><p id="par0015" class="elsevierStylePara elsevierViewall">She returned to the clinic 2 weeks later, with persistent symptoms and a fever of 38.5 °C, and was admitted to hospital with a primary diagnosis of complicated acute otitis media.</p><p id="par0020" class="elsevierStylePara elsevierViewall">A lumbar puncture (LP) was performed with increased opening pressure, leukocytosis with a predominance of mononuclear cells, and increased protein. Blood tests demonstrated leukocytosis with a predominance of neutrophils. Intravenous acyclovir and ceftriaxone were administered with symptomatic improvement. There was no bacterial or fungal growth in the cultures (blood culture, urine culture, and CSF culture), and the IgM titration for herpes virus was negative; consequently, the acyclovir was discontinued. Papillary oedema, a sign of endocranial hypertension, was visualised and acetazolamide was administered. She was discharged from hospital after 10 days with acetazolamide and meprednisone, with clear improvement of her facial paralysis and full recovery after one month.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Three months later, she consulted her paediatrician again for a new episode of holocranial headache and vomiting, together with isolated febrile spikes, similar to the previous episode. Symptomatic outpatient treatment with ibuprofen and metoclopramide was indicated.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Three weeks after that, she presented with persistence of symptoms. Physical examination revealed a stiff neck and signs of meningeal involvement, and it was therefore decided that she be admitted to the paediatric ward. In the clinical history, the otoscopic examination on admission referred only to tympanic congestion.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The multislice CT scan revealed mastoid involvement. A new lumbar puncture was performed, which showed increased protein and leukocytosis with a predominance of mononuclear cells. Treatment with ceftriaxone and acyclovir was started. The need to evaluate with nuclear magnetic resonance (NMR) without artefact was raised, which is why the ENT department was consulted, specifically the implanting physician. Until then, the paediatric department had not considered the possible otogenic origin of the problem. A medical meeting was held and it was decided that the otogenic origin of the problem caused by the cochlear implant’s internal processor magnet was otogenic, so that an MRI scan could be performed. During surgery, no signs of acute middle ear infection were detected; however, the eardrum exhibited a small atal tear, through which the electrode could be seen, a finding not previously reported in her clinical history.</p><p id="par0040" class="elsevierStylePara elsevierViewall">MRI with contrast revealed meningeal enhancement (a finding related to meningitis), associated with a hyperintense image in T2 in the internal auditory canal, up to the dural region with gadolinium uptake, compatible with an infectious process. The decision was made to request a consultation with the Otorhinolaryngology Service at the University of Munich in Germany, who agreed with the diagnosis and treatment instituted.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Because of this and given the slow evolution of the patient’s condition, it was decided that an audiological evaluation be performed, presenting vocal audiometry on the non-implanted side, with 100% discrimination at 70 dB. Due to the complication presented, the entire cochlear implant was removed, without complications, and was sent to bacteriology for culture. The intracochlear electrode bundle was not left in place as usual (due to a possible late reimplantation, so as to avoid fibrosis and intraluminal ossification) at the request of the infectious disease service to be able to study all of the prosthetic material and the possibility of <span class="elsevierStyleItalic">biofilm</span>.</p><p id="par0050" class="elsevierStylePara elsevierViewall">One week after removing the implant, a new MRI was performed, which showed a lesion in the left cerebellopontine angle with extension to the internal auditory canal, surrounding the seventh and eighth cranial nerves, hyperintense in T2 with gadolinium uptake, raising the possibility of a schwannoma of the eighth cranial nerve as a differential diagnosis. Small foci of internal necrosis and cerebral microabscesses were observed.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The new lumbar puncture was normal. The patient exhibited a favourable evolution of all her symptoms after surgery to remove the implant. The prosthesis underwent sonication and developed multisensitive <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span>. It should be noted that sonication is a procedure widely used in implantable trauma prostheses removed in cases of chronic infection.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The aim of this technique is to remove biofilm from prostheses using low-frequency and low-intensity ultrasound. The optimal ultrasound frequencies for sonication of <span class="elsevierStyleItalic">Staphylococcus aureus</span>, <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span>, and <span class="elsevierStyleItalic">Escherichia coli</span> are 35 and 40 kHz.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Medication was adjusted according to microbiological findings and post cochlear implant otogenic meningitis was diagnosed. Given the clinical improvement and laboratory parameters, the patient was discharged home with ceftazidime and metronidazole for 6 weeks. Evaluated in April 2020, the patient remained asymptomatic, with a new MRI scan showing a slight improvement compared to the previous ones.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0060" class="elsevierStylePara elsevierViewall">Otogenic meningoencephalitis is a rare post-surgical complication in implanted patients. It is well known that, as a prosthesis, the CI functions as a foreign body and can predispose to infections in patients with acute bacterial processes such as otitis media (FDA 2009).</p><p id="par0065" class="elsevierStylePara elsevierViewall">This patient used her implant for 6 years without complications until she had 2 recurrent episodes of AOM according to her medical records, which we believe is not correct. However, this description notwithstanding, when we discovered an atical perforation with the presence of an electrode bundle through it, we concluded that we were dealing with chronic otitis media with episodes of flare-ups, which were the origin of the meningeal symptoms.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Although the bacterial complication is expected to be due to pneumococcus<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> or <span class="elsevierStyleItalic">Haemophilus influenzae</span>, the presence of <span class="elsevierStyleItalic">Pseudomonas</span> in the culture leads us to consider colonisation through the tympanic perforation that the patient presented after the first flare-up of chronic otitis media.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Arndt et al. published a similar case of necrotising meningoencephalitis in the late postoperative period following cochlear implantation, associated with facial paralysis with a focus on the pontocerebellar angle.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The same work concentrates on the possibility of the similarity of the image in the internal auditory canal with neurinoma, which we ruled out on the basis of clinical and imaging evolution.</p><p id="par0080" class="elsevierStylePara elsevierViewall">In more than 1300 cases of cochlear implantation, Theunisse et al. mention only one case of post-implantation meningitis due to <span class="elsevierStyleItalic">Pseudomonas</span> as a late complication.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">We also highlight the importance of the ultrasound sonication technique to detach <span class="elsevierStyleItalic">biofilm</span> from the prosthesis in order to isolate the causal germ and provide appropriate antibiotic treatment. In conclusion, in the presence of any late complication apparently unrelated to the implant, such as facial paralysis, recurrent otitis media, and severe headaches, an imaging examination, especially with MRI, is mandatory, which is why the compatibility of these implants with MRI is also important.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interests" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-07-22" "fechaAceptado" => "2020-11-22" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Zernotti M, Filiberti G, Muller J, Zernotti M. Complicación tardía de implante coclear: meningoencefalitis necrosante. Acta Otorrinolaringol Esp. 2022;73:61–63.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Role of sonication in the microbiological diagnosis of implant-associated infections: beyond the orthopedic prosthesis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. Oliva" 1 => "P. Pavone" 2 => "A. D’Abramo" 3 => "M. Iannetta" 4 => "C.M. Mastroianni" 5 => "V. 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