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A rare case of primary otoscleroma of the middle ear

Published online by Cambridge University Press:  04 October 2012

A R Kakeri  and
A H Patel
A R Kakeri*
Affiliation:
Department of ENT, Al-Ameen Medical College, Bijapur, Karnataka, India
A H Patel
Affiliation:
Department of ENT, Al-Ameen Medical College, Bijapur, Karnataka, India
*
Address for correspondence: Dr Ashfak R Kakeri, Professor, Department of ENT, Al-Ameen Medical College Hospital, Bijapur 586108, Karnataka State, India E-mail: ashfak_kakeri@yahoo.com
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Abstract

Objective:

We report an extremely rare case of primary otoscleroma.

Method:

We present a case report and a review of the world literature concerning otoscleroma.

Results:

An adult woman presented with chronic suppurative otitis media with tubotympanic disease and conductive hearing loss. On mastoid exploration, dark granulations were seen, which were identified as otoscleroma on histopathological examination. The patient responded well to streptomycin.

Conclusion:

To the best of our knowledge, this is the first report of primary otoscleroma in the world literature. This case indicates that Frisch's bacillus can also spread to the middle ear.

Keywords

Ear, MiddleOtitis MediaRhinoscleroma

Information

Type
Clinical Records
Information
The Journal of Laryngology & Otology , Volume 126 , Issue 12 , December 2012 , pp. 1276 - 1277
Copyright
Copyright © JLO (1984) Limited 2012

Introduction

Rhinoscleroma or scleroma is a progressive, granulomatous disease which commences in the nose and which can eventually extend into the nasopharynx, oropharynx and larynx.Reference Weir, Golding-wood and Kerr1, Reference Kameshwaram2 It can sometimes affect the middle ear, hence the term otoscleroma. It is commonly seen in patients with poor hygieneReference Weir, Golding-wood and Kerr1 and in those of reproductive age.Reference Kameshwaram2–Reference Goravalingappa and Belagavi4 Frisch's bacillus has a predilection for the upper respiratory tract.Reference Kameshwaram2 It can also present at other associated sites, and with chronic suppurative otitis media with or without complications.Reference Barbary, Fouad and Fatt-Hi3

Here, we present a rare and interesting case of otoscleroma.

Case history

A 34-year-old woman presented to the out-patient department of Al-Ameen Medical College Hospital, Bijapur, with a 7-year history of left ear discharge, with reduced hearing over the previous 2 years.

Examination revealed a large central tympanic membrane perforation with polypoidal changes in the middle-ear mucosa.

An X-ray of the mastoid (Schüller's view) revealed sclerosis.

Her hemogram was normal.

Pure tone audiometry revealed a 40 dB, conductive hearing loss.

The patient underwent mastoid exploration under local anaesthesia. Intra-operatively, abundant dark, polypoid masses and granulations were found within the mastoid antrum and middle ear, from which tissue was obtained and sent for histopathological examination. The malleus and the lenticular process of the incus were eroded and necrosed. Granulations were present over the facial canal. The lateral semicircular canal dome was identified and found to be intact. There was a greater degree of peri-operative bleeding compared with routine cases. After removing the diseased tissue, a tragal cartilage ossiculoplasty (inter position) was performed together with a temporalis facial graft.

Post-operatively, cefotaxime (1 g) was administered intravenously twice daily for 5 days. The post-operative period was uneventful.

Histopathological examination (Figure 1) revealed chronic granulomatous changes with the presence of Mikulicz cells, Russell bodies and macrophages, indicating rhinoscleroma.

Fig. 1

Photomicrograph of otoscleroma showing macrophages, plasma Russell bodies and Mikulicz cells. (Hematoxylin–eosin stain ×200)

The patient's nose, paranasal sinuses, oral cavity and larynx were examined but no abnormality was detected. A biopsy was taken from the nasal mucosa and turbinates but did not reveal any scleromatous changes. Streptomycin (0.75 g) was administered intramuscularly (after a test dose) once daily for four weeks.

Three months after surgery, the graft had taken well, there was a 15 dB hearing improvement, and the mastoid cavity was dry.

Discussion

Rhinoscleroma per se involves the nose; however, it sometimes only affects the middle ear, in which case it is termed otoscleroma. The latter location is very rare, with only two previous reports.Reference Barbary, Fouad and Fatt-Hi3, Reference Goravalingappa and Belagavi4 Sites such as the lacrimal sac,Reference Badrawi6 larynx and trachea can also be affected.Reference Yasin5 Complications of otoscleroma have been reported: for example, Barbary et al. Reference Barbary, Fouad and Fatt-Hi3 reported otoscleroma with facial nerve palsy. However, in our case as well as that reported by Goravalingappa and Belagavi,Reference Goravalingappa and Belagavi4 there were no complications. Rhinoscleroma spreads along the submucosal plane from the nose into the nasopharynx, the eustachian tube and the middle ear. The larynx, trachea and lacrimal sacReference Weir, Golding-wood and Kerr1, Reference Barbary, Fouad and Fatt-Hi3–Reference Badrawi6 can also be involved.

  • • Primary otoscleroma is very rare; usually the nose is involved (rhinoscleroma)

  • • This adult case of primary otoscleroma presented with otorrhoea and deafness

  • • Mastoid exploration showed dark granulation tissue

  • • Diagnosis was made histologically from mastoid granulation tissue

  • • The patient responded to streptomycin

Occasionally, primary otoscleroma may be encountered without any evidence of disease elsewhere, i.e. the infective organism is inoculated directly into the middle ear,Reference Goravalingappa and Belagavi4 either by droplet infection from the nose or nasopharynx, or through the external ear via tympanic membrane perforation.

Various post-operative hearing outcomes have been reported. In our case, hearing improved by approximately 15 dB, whereas previously reported patients experienced no improvement in hearing.Reference Barbary, Fouad and Fatt-Hi3, Reference Goravalingappa and Belagavi4

References

1Weir, N, Golding-wood, DG. Infective rhinitis and sinusitis Rhinology. In: Kerr, AG, ed. Scott Brown's Otolaryngology, 6th edn.Butterworth–Heinemann: Oxford, 1997;4/8/34–35Google Scholar
2Kameshwaram, S. ENT Disorders in a Tropical Environment, 6th edn.MERF Publications: Chennai, 1999;39–52Google Scholar
3Barbary, A, Fouad, H, Fatt-Hi, A. Scleroma affecting the middle ear cavity with report of three cases. Ann Otol Rhinol Laryngol 1974;83:107–10CrossRefGoogle ScholarPubMed
4Goravalingappa, JP, Belagavi, CS. Scleroma in the middle ear: case report. Indian J Otolaryngol 1980;321:21Google Scholar
5Yasin, A. Primary scleroma of trachea. Gasette Kasr Aini 1958;24:3Google Scholar
6Badrawi, R. Dacryoscleroma. Ann Otol Rhinol Laryngol 1962;71:247–54Google Scholar
Figure 0

Fig. 1 Photomicrograph of otoscleroma showing macrophages, plasma Russell bodies and Mikulicz cells. (Hematoxylin–eosin stain ×200)