Chronic otorrhoea, otalgia and hearing loss not responding to antibiotics and tympanoplasty surgery

Abstract

Chronic otorrhoea from a tympanic membrane perforation is common. We present the case of a patient who had already received seemingly adequate treatment for his condition in the past. Yet, he presented to our outpatient clinic with worsening otalgia and otorrhoea, progressive hearing loss and a new tympanic membrane perforation. After a thorough otological evaluation, the patient’s medical history and the histological specimen from a previous operation were reviewed. The findings met the diagnostic criteria of eosinophilic otitis media. After treatment with topic triamcinolone through the perforated tympanic membrane, the patient’s otalgia subsided, hearing levels were improved and the size of the tympanic membrane perforation decreased.

Background

Otalgia, aural discharge and subacute hearing loss—separate or combined—are common causes for a presentation to a primary care physician or an ENT specialist. Topic or even systemic antibiotics may be indicated in case of infection. Surgery may be beneficial in persisting tympanic membrane perforation with recurring infections while it is mandatory in case of cholesteatoma. Intractable cases may warrant further workup, which may reveal eosinophilic otitis media (EOM).

Case presentation

A 49-year-old patient presented to our emergency department with a complaint of right-sided aural discharge, otalgia and slowly progressing hearing loss. Symptoms had started several months ago and were slowly deteriorating over the last few weeks. The patient noted that he had received surgery for similar symptoms 4 years ago. His symptoms had subsided for some years thereafter. His medical history was otherwise significant for bronchial asthma, which was poorly controlled lately.

Aural examination revealed brownish-yellow fluid in the right external ear canal (figure 1) draining from a small, central perforation in the postero-superior quadrant of the tympanic membrane (figure 2). A culture swab was obtained. The membrane itself was partially covered by the fluid and appeared slightly inflamed. While the contralateral otoscopy showed no alterations, endoscopic evaluation of the nasal cavity revealed second-degree polyposis in the middle meatus on both sides.

Figure 1.

Otoscopic evaluation on initial presentation shows highly viscous secretions near the tympanic membrane.

Figure 2.

After secretions are evacuated, a perforation is seen inside a slightly inflamed tympanic membrane.

With the working diagnosis of superinfected chronic suppurative otitis media (CSOM), topical antibiotic treatment was prescribed.

Investigations

Medical records revealed that the patient had received tympanoplasty surgery in CSOM with suspected cholesteatoma 4 years earlier. The operative report stated that cholesteatoma was ruled out, yet the middle ear had been partially occluded by eosinophilic, polypous formations intraoperatively. The intervention was nevertheless deemed successful, as reconstruction of the tympanic membrane was achieved, and follow-up visits had shown adequate healing at the time.

On his follow-up appointment in our clinic, 1 week after initial presentation, the patient’s symptoms had persisted. Aural examination showed a similar picture compared with the week before (figure 3). The swab culture had shown no growth of bacteria or fungi. Audiometric evaluation displayed a high-frequency, sensorineural hearing loss on the left, but a combined hearing loss across all frequencies on the right (figure 4).

Figure 3.

7 days after topical antibiotic treatment, the actual size of the tympanic membrane perforation can be demonstrated.

Figure 4.

Pre-treatment audiometry reveals normal bilateral bone-conduction thresholds up to 1 kHz with a subsequent drop to a severe hearing loss in higher frequencies. While hearing loss is completely sensorineural on the left, an air–bone gap of around 20 dB hearing level results in a combined mild to profound hearing loss on the right.

Further workup in our clinic consisted of aspiration of fluid/detritus from the patient’s middle ear with histological evaluation, which demonstrated eosinophilia (figure 5).

Figure 5.

Specimen from the patient’s middle ear demonstrates tissue formations dominated by eosinophilia among Charcot-Leyden crystals.

Differential diagnosis

Although its diagnostic criteria are well defined nowadays, EOM can still be considered a diagnosis of exclusion. The most common pathology in a case of chronic otorrhoea (originating from the middle ear), otalgia and associated hearing loss is CSOM. Cholesteatoma is less common in this triad of symptoms, yet it remains far more prevalent than EOM.

Since one of the minor diagnostic criteria of EOM is its non-responsiveness to conventional treatment, misdiagnosing it at first is not a failure per se—rather another step in the diagnostic ladder.

Treatment

Once the diagnosis of EOM was established, a literature review was performed. Following a treatment proposal from the leading research group of this entity, our therapy consisted of four sessions of intratympanic triamcinolone instillations with extrusion of the fluid through the eustachian tube with the help of a Politzer balloon. The patient rested with the head tilted to the contralateral side for 30 min after each session and was subsequently discharged. No adverse effects were observed.

Outcome and follow-up

The patient returned for regular follow-up visits consisting of clinical and audiometric evaluations. While otalgia and otorrhoea subsided, aural examinations demonstrated a partial closure of the previously noted tympanic membrane perforation (figure 6). The level of conductive hearing loss was significantly reduced after therapy (figure 7).

Figure 6.

Repeat otoscopy after 4 sessions of intratympanic triamcinolone shows a less inflamed tympanic membrane, no secretions and a slightly smaller perforation.

Figure 7.

Post-treatment audiometry demonstrates a significantly decreased air–bone gap. While hearing levels on the left are unchanged, the hearing loss on the right is now entirely sensorineural.

The patient has been in our clinic for follow-up once every other month for the past year. His complaints have recurred twice. However, intermittent injections of triamcinolone never failed to control his symptoms. We offered to send the patient for evaluation of antibody treatment, which he declined so far.

Discussion

Chronic otorrhoea is an extremely common symptom encountered in primary care or ENT departments.¹ The underlying pathology is normally correctly diagnosed and easily treated. Treatment can involve topical or systemic antibiotics and sometimes even surgery. In some cases, however, chronic otorrhoea can persist despite adequate therapy. It is then necessary to consider rare otological diseases like eosinophilic otitis—especially when polypous formations or viscous secretions arise from the middle ear or the patient suffers from comorbidities like asthma or nasal polyps.

The disease was first described and named accordingly in 1995 by a group of Japanese researchers.² It was not until 2002, however, that another group from Japan defined diagnostic criteria for the first time.³ In 2011, Iino et al performed a study on almost 140 patients with EOM with about the same number of age-matched controls. They then refined the previous definition, introducing major, minor and exclusion criteria.⁴ These criteria are well accepted nowadays and can effectively guide towards the correct diagnosis and treatment. According to this definition, EOM can be confidently diagnosed if the patient presents with a major and at least two minor criteria.

Diagnostic criteria of eosinophilic otitis media (EOM)

Major criteria:

• Otitis media with effusion or chronic otitis media with eosinophil-dominant effusion

Minor criteria:

• Highly viscous middle ear effusion

• Resistance to conventional treatment for otitis media

• Association with bronchial asthma

• Association with nasal polyposis

Exclusion criteria:

• Churg-Strauss syndrome

• Hypereosinophilic syndrome

To the best of our knowledge, no published statistics exist about the prevalence of EOM in any given population. The largest study populations only contained around 100 patients and seem to have found a preponderance for females in their sixth decade of life.⁵

Most studies covering this topic originate from Asia, specifically Japan. This may either hint towards a predominance in a certain ethnicity or could simply be explained by the fact that the first description of this entity came from this area.

Comorbidities like bronchial asthma and nasal polyposis are common. Asthma was prevalent in 90% of patients in the largest study covering this topic.⁴ While this analysis reported rhinosinusitis in 74% and nasal polyps in only 62% of patients, another article found it to be far more prevalent, reporting numbers as high as 87% for chronic rhinosinusitis.⁵

Multiple factors contribute to the pathophysiology of EOM. High concentrations of interleukin-5, ecalectin and eotaxin, all contributors to eosinophil attraction, were found in the middle ear effusions of affected patients.⁶ This most likely leads to the higher concentration of eosinophil cationic protein (ECP, a product of eosinophils), EG2-positive cells (an activated eosinophil marker) and eosinophils themselves.⁷

While eustachian tube dysfunction plays a decisive role in the pathogenesis of CSOM, patients with EOM seem to have a rather prolonged opening of the eustachian tube.⁸ This observation led the authors to conclude that “a patulous eustachian tube easily allows the entry of antigenic materials into the middle ear, causing eosinophil-dominant inflammation”. This theory illustrates the obvious association with bronchial asthma and the term ‘one airway, one disease’.

Early reports proposed systemic steroid therapy as the only effective treatment.³ Later, local steroid treatment through an existing perforation or via tympanostomy was introduced with significant success.⁹ Tympanostomy tube placement for continuous drainage was not recommended in this paper, as early blockage from the highly viscous secretions were frequently observed.

The role of surgery in EOM is yet to be determined. While early reports deem a surgical approach to be unsuccessful or ‘controversial’ at best, a new study describes myringoplasty as a favourable therapeutic option in severe cases.¹⁰

Lately, numerous studies have reported successful treatment of EOM with IL-5-antibody therapy.^11–13 This approach seems obvious, as these pharmaceuticals have been accepted in the therapy of asthma for a long time. It is therefore hardly surprising that a new paper described how the severity of EOM is closely related to the control of comorbid bronchial asthma. The authors strongly suggest adequate treatment of both conditions for an optimal outcome.¹⁴ They also suggest to escalate therapy according to the severity of the disease—from topic via systemic steroid through to surgery.

A chronic disease like EOM is rarely successfully treated after a short period. Hence, it is noteworthy to talk about long-term complications. Apart from chronic ear pain and disturbing otorrhoea, people suffering from EOM are likely to develop profound sensorineural hearing loss—a study from Japan reporting a prevalence of 47% in a study cohort, with an additional 6% of patients who were already deaf.¹⁵ Nakagawa et al reported that sensorineural hearing loss in EOM especially occurred at 4 kHz and 8 kHz and that the amount of hearing loss correlated significantly with the symptom duration before initiating treatment. Finally, reports about successful treatments of patients with EOM suffering from deafness with cochlear implantation exist.^{16 17}

While there are still no treatment guideline for EOM, the refinement of the disease’s classification over time and the evolution of different aspects of treatment demonstrate that we are currently on a journey from ‘steroids for all’ to patient-tailored therapy in the treatment of this otological disease—that for some should no longer be considered as ‘rare’.⁵

Learning points.

► Otorrhoea originating from the middle ear is mostly associated with chronic suppurative otitis media. Yet, if conventional treatment fails, additional investigations are indicated.

► A patient with (uncontrolled) bronchial asthma and nasal polyps suffering from a triad of otorrhoea, otalgia and hearing loss refractory to conventional therapy should be evaluated for eosinophilic otitis media (EOM).

► Steroid therapy (either local or systemic) and adequate control of a comorbid asthma condition represents a cornerstone in the treatment of patients with EOM. Monoclonal antibodies may represent a therapeutic option in non-responders. While surgery was controversially discussed for a long time, it now seems accepted in severe cases.

► Long-term complications in EOM are slowly progressing sensorineural hearing loss (most likely from toxic damage to the inner ear by inflammatory cytokines) and even deafness.