Laryngeal rhinosporidiosis: report of a rare case.

Abstract

Extranasal manifestations of rhinosporidiosis are relatively uncommon. Laryngeal involvement is extremely rare, as only 3 cases have been previously reported. We describe a new case, which occurred in a patient with coexisting nasal rhinosporidiosis who presented with inspiratory stridor. Both lesions were completely excised under general anesthesia without the need for preliminary tracheostomy.

Introduction

Rhinosporidiosis in the larynx is rare. In this article, we describe what we believe is only the fourth such case, all of which have occurred in India.

Case report

A 56-year-old woman from northwestern India presented with a 4-month history of progressively increasing noisy breathing. The patient had been diagnosed with nasal rhinosporidiosis 12 years earlier, and she had undergone nasal surgery three times to treat recurrences.

On examination, she was noted to have inspiratory stridor. Indirect laryngoscopy revealed that a pedunculated mass had arisen from the right ventricular band; the mass moved in and out of the glottic opening with respiration. Anterior rhinoscopy revealed that multiple irregular masses had filled both nasal cavities. Flexible nasopharyngolaryngoscopy confirmed the findings of the indirect laryngoscopy (figure 1). Radiologic examination of the cervical region delineated a mass in the supraglottic region. There was no narrowing of the subglottic airway.

[FIGURE 1 OMITTED]

An excision of the laryngeal and nasal masses by carbon dioxide (C[O.sub.2]) laser was planned. The oral cavity and posterior pharyngeal wall were sprayed with 10% lignocaine (lidocaine). Under fiberoptic laryngobronchoscopic guidance, intubation was achieved with a 5-mm uncuffed endotracheal tube. Anesthesia was maintained with intravenous fentanyl and propofol. The endotracheal tube was then replaced by a 1.4-mm-diameter metal cannula past the mass, and oxygen was injected via a jet ventilation system. Atracurium was used to paralyze the patient and to aid in ventilation. A Storz laryngoscope was passed along the ventilation cannula, and the laryngeal mass was excised completely at the pedicle with the C[O.sub.2] laser. The metal cannula and the laryngoscope were removed. A 6.5-mm endotracheal tube was introduced for continuation of anesthesia, and the nasal masses were then excised.

The patient's postoperative course was uneventful, and she was started on dapsone for prevention of recurrence. Histopathologic examination revealed the presence of multiple sporangia that contained spores of Rhinosporidium seeberi (figure 2).

[FIGURE 2 OMITTED]

Discussion

R seeberi (Rhinosporidium kinealyi) is a member of the Phycomycetes class of fungi. Invasion by this fungus is universal, but it is endemic in India and Sri Lanka. The first known case of rhinosporidiosis was identified in 1892 by Malbran of Buenos Aires. (1) Seeber published the first detailed account of rhinosporidiosis in 1900. (2) In 1923, Ashworth (3) described the fungus' life cycle, and in 1964, Karunaratne published a complete review of rhinosporidiosis in man. (4)

Rhinosporidiosis predominantly affects the mucous membranes of the nose and nasopharynx; it also occasionally involves the lips, palate, uvula, maxillary antrum, conjunctiva, lacrimal sac, epiglottis, larynx, trachea, bronchus, ear, scalp, skin, penis, vulva, and vagina. (5)

The mainstay of treatment is surgical excision by laser or electric diathermy. Medical treatment with dapsone has been recommended for the prevention of recurrence. (6)

In our review of the literature, we found only 3 reports of laryngeal rhinosporidiosis, all of which had occurred in India. (7,8) In the first report, published in 1974, Pillai described 2 patients who presented with nasal and laryngeal masses. (7) Both were excised under local anesthesia. In 1996, Banerjee et al reported the case of a patient who presented with breathing difficulty and a history of nasal surgery. (8) Following a preliminary tracheostomy, the laryngeal mass was excised trader general anesthesia via an external approach.

By using current general anesthesia techniques on our patient, we were able to avoid a tracheostomy and remove both the laryngeal and nasal lesions during the same procedure.

To the best of our knowledge, the clinical presentation of laryngeal rhinosporidiosis with inspiratory stridor has not been previously reported.

References

(1.) Kameswaran S, Lakshmanan M. Rhinosporidiosis. In: Kameswaran S, Lakshmanan M, eds. ENT Disorders in a Tropical Environment. Chennai, India: MERF Publications, 1999:19-34.

(2.) Seeber GR. Un nuevo esporozoario parasito del hombre: Dos casos encontrades en polipas nasales [thesis]. Buenos Aires: Universidad Nacional de Buenos Aires, 1900

(3.) Ashworth JH. Rhinosporidium seeberi (Wernicke, 1903) with special reference to its sporulation and affinities. Trans R Soc Edinburgh 1923;53:301-42.

(4.) Karunaratne WA. Rhinosporidiosis in Man. London: Athlone Press, 1964.

(5.) Kerr AG, ed. Scott-Brown's Otolaryngology. Vol. 4. Oxford: Butterworth-Heinemann. 1997.

(6.) Job A, Venkateswaran S, Mathan M, et al. Medical therapy of rhinosporidiosis with dapsone. J Laryngol Otol 1993;107:809-12.

(7.) Pillai OS. Rhinosporidiosis of the larynx. J Laryngol Otol 1974;88: 277-80.

(8.) Banerjee SB, Sarkar A, Mukherjee S, Bhownik A. Laryngeal rhinosporidiosis. J Indian Med Assoc 1996;94:148, 150.

From the Department of Otolaryngology--Head and Neck Surgery (Dr. Kumar, Dr. Mathew, and Dr. Kurien), the Department of Anaesthesia (Dr. Cherian), and the Department of Pathology (Dr. Rozario), Christian Medical College, Vellore, Tamilnadu, India.

Reprint requests: Dr. John Mathew, Professor, Department of Otolaryngology--Head and Neck Surgery, Christian Medical College, Vellore 632 004, Tamilnadu. India. Phone: [phone] or 91-416-521-2798; fax: [phone] or 2035: e mail: [e-mail]