Subtotal petrosectomy and cochlear implantation for severe eosinophilic otitis media with infection: A case report

Abstract

Eosinophilic otitis media (EOM) is an intractable otitis media characterized by highly viscous secretions containing eosinophils in the middle ear. Clinically, bacterial infection complicates the course of EOM and may accelerate the progression of sensorineural hearing loss. We present a case of a woman in her late 50s, diagnosed with severe EOM, experienced recurrent ear infections despite repeated tympanostomy tube insertions, intratympanic corticosteroid injections, and systemic corticosteroid treatment. Microbiological analysis of middle ear effusion revealed methicillin-resistant Staphylococcus aureus and Candida parapsilosis coinfection. The use of corticosteroid and biologic agents was contraindicated due to the active infectious process. The patient underwent surgical debridement by subtotal petrosectomy, and obtained a successful infection control. Following resolution of the infectious process, the patient with bilateral severe mixed hearing loss and inadequate benefit from conventional hearing aids underwent successful cochlear implantation (CI), achieving satisfactory auditory rehabilitation. In cases of severe EOM complicated by refractory infections, subtotal petrosectomy represents a potentially effective therapeutic strategy for infection control and disease progression mitigation. Subsequent CI may be considered as a viable option for auditory rehabilitation in selected cases.

Introduction

Eosinophilic otitis media (EOM) is a challenging and distinct form of intractable otitis media. Diagnostic criteria were established in 2011. ¹ The clinical presentation of EOM typically features middle ear effusion rich in eosinophils and progressive hearing loss, frequently associated with comorbid conditions including bronchial asthma and chronic rhinosinusitis with nasal polyposis. ² Current therapeutic strategies primarily encompass corticosteroid, antibiotics, and surgical interventions including myringotomy with tympanostomy tube placement. ³ Emerging biologic therapies, mainly monoclonal antibodies targeting type 2 inflammatory pathways, have recently gained attention as potential therapeutic options for refractory EOM cases. ⁴ However, the evaluation of treatment efficacy remains challenging due to limited high-quality evidence and heterogeneity in clinical presentation. Specially, the management of severe EOM complicated by refractory infections represents a significant therapeutic challenge in clinical practice. While corticosteroid therapy (both systemic and topical) remains the cornerstone of EOM management, its use is associated with increased infection risk, potentially exacerbating middle ear mucosal pathology. This risk is particularly pronounced in severe cases characterized by granulation tissue formation within the tympanic cavity, which creates an environment conducive to microbial proliferation. Furthermore, the dual inflammatory processes involving both eosinophilic and neutrophilic pathways contribute to progressive sensorineural hearing loss (SNHL), ⁵ significantly impacting patients’ quality of life. This case report presents the therapeutic challenges encountered in managing a severe EOM case complicated by methicillin-resistant Staphylococcus aureus (MRSA) and fungal coinfection.

Case report

This case report was prepared in accordance with the CARE guidelines for case reports. ⁶ A woman in her late 50s presented with a nine-year history of bilateral aural fullness, otorrhea, and progressive hearing loss, initially conductive but later mixed. Initial pure-tone audiometry (PTA) performed seven years prior to presentation demonstrated bilateral air-conduction (AC) thresholds of 51 dBHL, with bone-conduction (BC) thresholds of 17.5 dBHL (Figure 2(a)). She underwent a bilateral myringotomy with tube insertion, but the tubes became blocked by sticky effusion within one week. Subsequently, she received intratympanic injections of triamcinolone acetonide (40 mg/mL, 1 mL per injection) administered three times at monthly intervals over three months, which provided only temporary relief before recurrent middle ear effusions developed. Systemic corticosteroids (prednisone acetate 60 mg once daily for one week) were effective but limited by potential severe side effects. Three years ago, the AC hearing threshold in both ears had deteriorated to 100 dBHL, and the BC hearing threshold was 60 dBHL (Figure 2(a)). Her medical history was significant for hypertension, coronary artery disease, and Samter's triad (asthma, nasal polyposis, and aspirin-exacerbated respiratory disease), with three prior endoscopic sinus surgeries.

Five months prior to admission, she developed bilateral purulent otorrhea, with microbiological cultures identifying MRSA and Candida parapsilosis coinfection. Cytological analysis of middle ear effusion demonstrated 3% eosinophils and 97% neutrophils, while peripheral blood analysis revealed 13.1% eosinophilia and elevated serum IgE levels (409 KU/L). Otoscopic examination revealed granulation tissue filling both EACs and the tympanic cavity (Figure 1(a)), along with eosinophil-dominant exudate with plasmacytes and lymphocytes. PTA confirmed severe mixed hearing loss bilaterally (Figure 2(a)). A bone conduction hearing aid (BCHA) was fitted for the right ear, with an average hearing threshold of 60 dBHL and a speech recognition rate of 65% for bisyllable words (Figure 2(b)). Temporal CT showed bilateral diploic mastoids and soft tissue surrounding the ossicles with blurred margins (Figure 2(d)). She was diagnosed with severe EOM.

Figure 1.

Otoscopic and microscopic examinations. (a) The tympanic mucosa was covered with granulation tissue in the left ear. (b) Microscopic examination showed lots of granulations and sticky discharge in the tympanic and mastoid cavity during Stage I surgery. (c) The left tympani was well-epithelialized seven months after Stage I surgery. (d) At Stage II surgery, some viscous secretion in the deep tympanic cavity. (e) Postoperative cavity following subtotal petrosectomy of the right ear. The surgical cavity demonstrates complete removal of air cells, including those surrounding the labyrinth and internal carotid artery. (f) Twenty months after Stage II surgery, the epithelialized tympanic surface bulged a small bag in the right ear.

Figure 2.

Patient's hearing, histological and radiographic examinations. (a) Progression of hearing loss in both ears, showing changes in AC, BC, and ABG thresholds over time. The patient initially presented with moderate conductive hearing loss, which progressed to severe mixed hearing loss. (b) Hearing outcomes with assistive devices before and after CI. Hearing thresholds across frequencies (0.25–6 kHz) and speech recognition rates are shown. Preoperatively, the patient's average hearing threshold with BCHA was 60 dBHL, with a bisyllabic word recognition rate of 65% at 70 dB SPL. At two months post-CI, the hearing threshold improved to 40 dBHL, with no change in bisyllabic word recognition rate (65% at 70 dB SPL). By 20 months post-CI, the hearing threshold remained at 40 dBHL, with a bisyllabic word recognition rate of 53% at 65 dB SPL and a short-sentence recognition rate of 70%. (c) The histological examination of the granulation tissue collected from the tympanic cavity showed eosinophil-dominant inflammatory exudate with plasmacytic and lymphocytes (hematoxylin and eosin staining, *200). (d) Temporal CT showed bilateral diploic mastoids and ossicles surrounded by soft tissue with rough edges before surgery. (e) Temporal CT showed a well-defined left middle ear cavity except the antrum and a right middle ear filled with soft tissue density ten months after Stage I surgery.

The patient was admitted to the Department of Otolaryngology-Head and Neck Surgery at Peking Union Medical College Hospital (Beijing, China) in May 2021. Patient consent to treatment was obtained. Due to refractory infection, she underwent a subtotal petrosectomy for debridement. Intraoperative exploration revealed a large perforation of the tympanic membrane, granulation tissue, and viscous discharge filling most air cells (Figure 1(b)), including those around the labyrinth and internal carotid artery. All air cells were cleared, the cartilaginous portion of the Eustachian tube was cauterized, and a temporal muscle graft sealed the orifice. The lower mastoid cavity was obliterated using a subcutaneous muscle flap from the occipital region. The histological examination showed eosinophil-dominant inflammatory exudate with plasmacytic and lymphocytes (Figure 2(c)).

Postoperative follow-up at seven months demonstrated complete epithelialization of the tympanic cavity on otoscopic examination (Figure 1(c)), with temporal CT showing localized soft tissue density within the cavity (Figure 2(e)). Secondary-stage surgery involving cochlear implantation (CI) was performed 10 months following the initial procedure. Residual mucoid secretions in the deep tympanic cavity (Figure 1(d)) were meticulously removed. Electrodes (Cochlear Ltd, Australia) were inserted, the surgical cavity was filled with abdominal fat, and a dermal fat flap was used to seal the external site. And subtotal petrosectomy was performed in right (Figure 1(e)). Two months after surgery, hearing improved with a 40 dBHL average threshold and 65% speech recognition for bisyllable words. Long-term follow-up at 20 months post-stage II surgery revealed sustained improvement in aided hearing thresholds, with speech recognition scores of 53% for bisyllable words and 70% for short sentences in the left ear (Figure 2(b)). No recurrence of effusion was observed, although a small bulge of epithelialized tympanic tissue was observed in the right ear (Figure 1(f)).

Discussion

This study presents a unique case of bilateral severe EOM complicated by coinfection with MRSA and fungal pathogens. Such a complex case offers valuable insights into the management of EOM, which is often challenging due to its intractable nature and potential for hearing impairment.

The necessity of surgical treatment for infection in EOM

The management of EOM requires individualized treatment strategies tailored to disease severity and clinical presentation. ² Patients with mild disease may respond to conservative measures, while severe cases often necessitate aggressive interventions combining surgical and biologic therapies.^2,4,7 Infections associated with EOM are not infrequent. ⁵ The presence of eosinophils and neutrophils in the inflammatory response during EOM-related infections increases the risk of SNHL. ⁸ In this case, despite the used of both local and systemic antibiotics, the deep-seated bacterial colonization within granulation tissue proved refractory to eradication. The prolonged corticosteroid therapy required for EOM management paradoxically increased susceptibility to secondary fungal infections, significantly complicating the therapeutic course. Consequently, conventional medical management strategies, including corticosteroid and antimicrobial therapies, proved insufficient for disease control. Therefore, surgical intervention becomes imperative to disrupt the persistent cycle of infection and inflammation, a critical step in preventing further auditory deterioration and improving clinical outcomes.

The superiority of subtotal petrosectomy

In cases of EOM complicated by refractory infections, conventional mastoidectomy often proves inadequate for complete disease control. This limitation stems from its inability to address the persistent inflammatory source in the Eustachian tube, which serves as a conduit for eosinophilic inflammation reflux into the middle ear cavity. ⁹ In contrast, subtotal petrosectomy has emerged as the surgical intervention of choice for refractory otitis media, offering superior outcomes in selected cases. ¹⁰ This surgical approach achieves complete Eustachian tube obliteration, effectively preventing the retrograde flow of eosinophilic inflammation from the nasopharynx to the middle ear cavity. In the present case, subtotal petrosectomy successfully achieved complete removal of granulation tissue and viscous secretions from the tympanic and mastoid cavities, including peri-labyrinthine and peri-carotid regions. However, postoperative CT and findings during the second-stage surgery revealed residual mucoid secretions in the left tympanic cavity, and a bulging epithelialized mass was observed in the right tympanic membrane. These observations suggest that inflammation from the petrous apex air cells may spread to the middle ear through the infra-labyrinthine region. However, this hypothesis requires further validation with larger case series. Despite these limitations, subtotal petrosectomy remains a critical intervention for controlling infection and creating a relatively stable environment for subsequent auditory rehabilitation in complex EOM cases.

Hearing rehabilitation and CI

After controlling the infection through subtotal petrosectomy, auditory rehabilitation emerged as the primary therapeutic focus. The patient suffered from profound mixed hearing loss, with eosinophilic-mediated damage to the ossicular chain and cochlea. ¹¹ Although a previous case report suggested that BCHA could improve hearing in patients with EOM, the specific outcomes were not detailed, and the benefit is presumed to be limited. ¹² In our case, the BCHA provided inadequate auditory benefit, particularly in high-frequency hearing, likely due to the inherent limitations of BCHA technology and persistent cochlear damage. CI, which directly stimulate the spiral ganglia, are effective in improving SNHL caused by eosinophilic inflammation. ¹³ In this case, despite preserved residual hearing, the suboptimal performance of the BCHA necessitated alternative rehabilitative strategies. Although there is no direct evidence that EOM causes cochlear ossification, a single case report has suggested that early CI implantation may prevent the loss of implantation opportunities due to potential cochlear ossification. ¹⁴ Therefore, early CI was considered crucial in this patient. It is important to note that before CI surgery, ensuring the patient's ear meets suitable implantation conditions is of utmost importance. Residual high air cells or epithelium in the surgical cavity can increase the risk of postoperative recurrence, potentially leading to CI failure and the need for secondary surgery to remove the device. Postoperatively, the patient's aided hearing thresholds remained stable, but speech recognition rates did not significantly improve. The outcomes of CI implantation are influenced by multiple factors, including the duration of deafness, preoperative speech perception abilities, rehabilitation training, and psychological support. ¹⁵ However, the possibility of progressive eosinophilic inflammation affecting the cochlea and retro-cochlear structures cannot be excluded. ¹⁴ These findings provide valuable insights for managing similar complex cases and highlight the need for further research into the long-term effects of eosinophilic inflammation on CI outcomes.

Research value and limitations

This case presents a classic example of severe EOM refractory to conventional corticosteroid therapy, which was poorly tolerated and ineffective in controlling disease progression. The patient's recurrent infections and persistent otorrhea necessitated an aggressive surgical approach. Subtotal petrosectomy provided rapid and effective relief of otorrhea, and created a relatively stable environment for subsequent CI. Notably, biologic agents were not utilized in this case due to the patient's active infections, personal preference and the lack of robust evidence supporting their efficacy in such complex cases. This underscores the need for further research into the role of biologics in EOM management. While this case provides valuable insights into the management of refractory EOM, it is important to acknowledge its limitations as a single case report. The findings should be interpreted with caution, and further studies with larger case series are urgently needed to validate and optimize these approaches.

Conclusion

In patients with severe EOM complicated by refractory infections, subtotal petrosectomy represents an effective surgical intervention for infection control and disease progression mitigation. CI may be considered as a viable auditory rehabilitation option following complete air cell clearance and Eustachian tube orifice closure, particularly in cases where BCHAs provide inadequate benefit.

Supplemental Material

sj-pdf-1-sci-10.1177_00368504251333048 - Supplemental material for Subtotal petrosectomy and cochlear implantation for severe eosinophilic otitis media with infection: A case report

Supplemental material, sj-pdf-1-sci-10.1177_00368504251333048 for Subtotal petrosectomy and cochlear implantation for severe eosinophilic otitis media with infection: A case report by Wanru Zheng, Xi Wang, Xu Tian, Zhen Huo, Zhiqiang Gao and Guodong Feng in Science Progress