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Disseminated cutaneous rhinosporidiosis in a HIV sero - positive patient
2 Departments of Oto-Rhino -Laryngology, Rajah Muthiah Medical College, Annamalai University, Annamalai Nagar - 608002. TN, India
Correspondence Address:
L Padmavathy
B3, R.5.A.Complex, Annamalai University, Annamalai Nagar - 608002. TN
India
How to cite this article: Padmavathy L, Rao I L, Selvam SS, Sahoo C G. Disseminated cutaneous rhinosporidiosis in a HIV sero - positive patient. Indian J Dermatol Venereol Leprol 2001;67:332-333 |
Abstract
Rhinosporidiosis is a chronic disease, classically involving the nose and nasopharynx, clinically presenting as polypii. However,disseminated rhinosporidiosis, involving various other sites including lips, conjunctiva, uvula, vagina, larynx. trachea. scalp and skin is also known to occur. A case of rhinosporidiosis, with nasal, oropharyngeal and multiple cutaneous lesions, in a patient who is sera - positivafor HIV is reported.Introduction
Rhinosporidiosis is a chronic, polypoid granulomatous disease of the anterior nares and nasopharynx.[1] It is caused by the fungus Rhinosporidium seeberi. Uncommonly other sites also can be affected like the skin, ocular, oral and genital mucous membranes. We report a case of disseminated cutaneous rhinosporidiosis in a 29-year- old man carpenter, who was HIV sero - positive.
Case Report
A 29 -year -old man, carpenter by occupation presented with the complaint of a progressively increasing nasal swelling which was causing obstruction for 4 months. He had mucoid nasal discharge and epistaxis for the same period. He also had change of voice, but had no dysphagia, Anterior and posterior rhinoscopy revealed an erythematous granular, polypoid growth in the left nostril, and nasopharynx protruding into the oral cavity while displacing the soft palate. The mass was clinically very suggestive of rhinosporidiosis. The uvula was edematous. He also had multiple non - tender warty lesions on body of 4 months duration.
Cutaneous examination showed multiple, hyperkeratotic,verrucous, non-tender papules on the trunk, limbs and face. He did not have any other systemic abnormalities. Hematological, including VDRL test, and biochemical investigation results were within normal limits. However, he was HIV positive by both tridot and Elisa methods. An X - ray of the paranasal sinuses revealed left maxillary haziness.
On detailed questioning, he admitted to having had multiple heterosexual contacts with many partners. There was no evidence of any other STDs clinically. A skin biopsy from two lesions, one from the trunk and another from the face, showed typical histopathological features of cutaneous rhinosporidiosis i.e. fibromyxomatous stroma with vascularity throughout, in the sections studied. Round cells containing sporangia were also seen.
The patient absconded before surgical excision of the nasopharyngeal mass could be undertaken. For the same reason, other medical modalities of therapy could not be tried.
Though seropositive for HIV, there was no other evidence of immunodeficiency, clinically or by the other available parameters.
Discussion
Rhinosporidiosis is a chronic granulomatous disease of the mucocutaneous tissue of man and animals caused by Rhinosporidium seeberi. The fungus belongs to the class chytidomycetes and the order chytidialis.
The disease is characterised by the production of large polypoid, tumor-like, papillomatous or warty lesions, which are hyperplastic, highly vascularised and friable. They may be sessile or pedunculated.
The organism has not been grown on culture nor has ever been successfully transmitted to experimental animals. Seeber, described the unique round structures histologically diagnostic of rhinosporidiosis as a protozoan.[2]
Ashworth, later described it as sporangium of a fungus and designated it as Rhinosporidium seeberi.[3] Ahluwalia, recently described these as lysosomal bodies loaded with indigestible residue some what reminescent of lysosomal storage diseases.[4] Therefore, ′sporangia′ have been redesignated as nodular bodies and `spores′ as spheres of cellular waste.
Immunohistochemical and electron microscopic studies showed transepithelial elimination of nodular bodies and destruction of some late stage nodular bodies in "histiocytic granulomata with central neutrophilic microabscesses.[1] Early nodular bodies were immurohistochemically positive for alpha- AT, alpha - ACT, CEA, S100, fibronectein, amyloid - p component, IgA, IgG, CIq, and C3.
The process of trans-epithelial elimination in this condition appears to be a significant but unsuccessful biological phenomenon of the host, attempting to clear the lesion and control the inflammatory processes. However this mechanism is not totally effective. No such changes could be demonstrated histologically in our case. Due to lack of facilities E.M, study could not be undertaken.
Rhinosporidiosis remains a chronic inflammatory disease with no medical treatment and only amenable to surgical excision with risks of recurrence and occasional wide spread and fatal disseminations.[5]
Remission of infections with dapsone[6] and ketoconazole[7] are described. However, no definitive therapy could be instituted in our patient as he absconded.
Rhinosporidiosis has been reported from many parts of the world on rare occasions but is endemic in India and Sri Lanka. Our patient seems to have a disseminated type of disease, associated with HIV seropositive state, which is not very common. However, he had no other evidence of immunodeficiency clinically. In the present case, whether immunocompromised status is the cause of dissemination, is not clear, as the patient did not undergo complete investigation. For the same reason, the response to treatment also could not be seen.
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