Case Report Urol Int 1992;49:224-226
Departments of Genitourinary Surgery and Pathology, Cancer Institute (WIA), Adyar, Madras, India
Rhinosporidiosis Mimicking Penile Malignancy
K e y w o rd s
A b stra ct
Penis Penile neoplasms Rhinosporidiosis
Rhinosporidiosis is a chronic infection which is endemic in India and Sri Lan ka. Penile involvement in rhinosporidiosis is rare and we report one such case. The pathology, treatment and possible etiology of the condition are re viewed.
Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, an endosporulating organism that is usually classified among fungi [1], The disease is endemic in South India and Sri Lanka, 9 out of every 10 cases reported in the world literature being from this region [2, 3], The lesion usually presents as a friable, pedunculated or sessile, polypoid mass involving mainly the nasal mu cosa in 70% of the cases [2], The less common sites of involvement include the nasopharynx, maxillary antrum, conjuctiva, tracheobronchial tree, larynx, skin, vagina, vulva and rectum [4, 5], Rarely, involvement of bone has been described [6], The lesion is rarely seen in western countries especially if located outside the nasal cavity. Penile involvement in rhinosporidiosis is a rare occur rence and herein we report one such case.
Case Report A 55-year-old male presented to the urology services with a 10year history of painless, polypoid lesions over the glans and external urethral meatus. The lesions were first seen over the external meatus but gradually spread to involve the glans penis. The lesions occasion ally bled and fell off but never regressed completely. The lesions were excised elsewhere 6 years ago but recurred and he was referred to our
Received: November 19. 1991 Accepted after revision: March 10. 1992
centre as a case of penile malignancy. The patient used to bathe regu larly in a temple pond frequented by many people. On examination, multiple polypoid and vascular lesions measur ing about 2.0 cm were seen arising from the urethra over the external meatus and fossa navicularis. In addition, the entire surface of glans was studded with smaller lesions (fig. 1). A biopsy of the lesion was done. Histopathology showed edematous fibrovascular tissue lined by hyperplastic squamous epithelium. The subcpithelial tissue showed a large number of sporangia measuring 10-200 pm and con taining hundreds of endospores (fig. 2). Some of the sporangia had ruptured and endospores were seen in the surrounding tissue. A diag nosis of rhinosporidiosis was made. In view of the extensive involve ment of the glans, a partial amputation of penis was carried out. The patient is doing well 6 months postoperatively without any recur rence.
D iscussion
Rhinosporidiosis of the penis is a rare lesion. It was first reported by Ingram [7] in 1910 and since then about 37 cases have been reported in the English literature [810]. The disease manifests as urethral involvement usually restricted to the fossa navicularis and external ure thral meatus. The lesions are seen as friable, vascular, polypoid or sessile masses arising from the terminal ure thra. Involvement of the glans is unusual and extensive involvement can mimic a penile neoplasm [7],
Dr. R. Ravi. Assistant Professor Department of Genitourinary Surgery Division of Surgical Oncology Cancer Institute (WIA) Adyar. Madras 600 020 (India)
© 1992 S. Kargcr AG. Basel 0042-1138/92/0494-0224 $2.75/0
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R. Ravia V.S. Mallikarjunab H.K. Chaturvedia
Fig. 2. Mature sporangium of R. seeberi containing endosporcs seen in the stroma of the penile lesion along with trophocytes in var ious stages of development. HE X 250.
The disease is caused by Rhinosporidium seeberi, an organism first described and identified as a protozoan by Seeber [ 11 ] in 1900. Ashworth [ 12] in 1923 suggested it to be a fungus and it is classified under phycomycetes although its fungal nature has been disputed [13]. Al though earlier efforts to culture the organisms [8, 14, 15] or transmit them to laboratory animals [2, 16] were unsuccessful, recently the organisms have been cultured by Krishnamoorthy et al. [17]. These authors found posi tive cultures in 92% (23 of 25) of their cases and demon strated it to be a fungus because of the presence of conidium. In its life cycle, the early trophocyte is about the size of a red blood cell, covered by chitin and lies freely in the con nective tissue. It gradually increases in size and the wall thickens. At the size of 45 pm diameter, the nucleus begins to divide. By the 14th division, the trophocyte becomes 140 pm in diameter and contains 16,000 young spores, each measuring 3 pm in diameter. Maturation of the spores occurs with the formation of mucoid envelope with chitinoid film, enlargement to 7-9 pm and the appearance of refringent spherules, 10-16 in number. The mature spo rangium is about 200-250 pm in diameter and it ruptures to release the spores through the germinal pore. Although the exact mode of transmission has not been clearly established, it is likely that contaminated water
and soil is the source of infection. This is strengthened by the observation that the disease is more common in males, particularly from rural areas, who have a habit of swimming in stagnant water. The disease is also found in a variety of animals like horses, mules, cattle and dogs and could possibly be transmitted to humans by contami nated water. Samaddar and Sen [5] found that more than 90% of their 166 patients with nasal rhinosporidiosis had the habit of bathing in ponds frequented by animals. The question of sexual transmission has been raised by the description by Symmers [ 18] of vulval rhinosporidiosis in a female and penile urethral rhinosporidiosis in a male, both of whom were sexual partners. However, similar evi dence of sexual transmission or sexual aberrations in patients with penile rhinosporidiosis has not been found by others [10]. Medical treatment in the form of trivalent and pentavalent antimony compounds has been tried and found to be of little value. Surgical excision of the lesion(s) is the treatment of choice. Partial amputation may be necessary if there is diffuse involvement of the glans as well. Recur rences are seen in up to 25% of patients with urethral rhi nosporidiosis [10], However, its incidence can be de creased considerably by the use of a diathermy knife for excision and electrocauterisation of the base of the le sion.
225
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Fig. 1. Multiple polypoid lesions are seen arising from the exter nal urethral meatus and fossa navicularis. The tip of the glans is swollen and covered with minute lesions. The probe is in the ure thra.
1 Friedman I, Osborn DA: Pathology of Granu lomas and Neoplasms of the Nose and Para nasal Sinuses. Edinburgh. Churchill Living stone, 1982, pp 1-64. 2 Karunaratne WAE: Rhinosporidiosis in Man. London, Althone Press, 1964, p 21. 3 Kameswaran S: Surgery in rhinosporidiosis. Experience with 293 cases. Int J Surg 1966;46: 602-605. 4 Mohapatra LN: Rhinosporidiosis; in Baker RD (ed): Human Infection with Fungi, Actinomy cètes and Algae. Berlin, Springer, 1971, chap 14, pp 676-683. 5 Samaddar RR, Sen MK: Rhinosporidiosis in Bankura. Indian J Pathol Microbiol 1990:33: 129-136. 6 Aravindan KP. Viswanathan MK, Jose L: Rhi nosporidiosis of bone. Case report. Indian J Pathol Microbiol 1989;32:312-313. 7 Ingram AC: Rhinosporidium kinealyi in un usual situations. Lancet 1910;ii:726.
226
8 Dhayagude RC: Unusual rhinosporidial infection in man. Indian Med Gazette 1941 ;76:513— 515. 9 Palaniswamy R, Bhandari M: Rhinosporidio sis of male terminal urethra. J Urol 1983:129: 598-599. 10 Sasidharan K, Subramanian P, Moni VN, Aravindan KP, Chally R: Urethral rhinosporidiosis. Analysis of 27 cases. Br J Urol 1987; 59:66-69. 11 Seeber GR: Un nueva esporonario parasite del hombre. Dos casos encontractos an polipos nasales. Jesis, Univ Mac de Buenos Aires, 1900, pp 1-620. 12 Ashworth JH: On Rhinosporidium seeheri (Wernicke, 1903), with special reference to its sporulation and affinities. Trans R Soc Edin burgh 1923;53:301-342. 13 Vanbreuseghem R, Devroy CH, Takashio M: Practical Guide to Medical and Veterinary' My cology. New York, Masson, 1978, p 227.
14 Rajan RV, Viswanathan GG, Rao AR, Rangiah PN, Anguli VC: Rhinosporidiosis: A study with report of a fatal case of systemic dissemi nation. Ind J Surg 1955;17:269-298. 15 Satyanarayana C: Rhinosporidiosis with a rec ord of 255 cases. Acta Otolaryngol 1960;51: 348-366. 16 Reddy DG, Lakshminarayana CS: Investiga tion into transmission, growth and serology in rhinosporidiosis. Indian J Med Res 1962;50: 363-369. 17 Krishnamoorthy S, Seerdharan VP. Koshy P. Kumar S, Anilkumari CK: Culture of Rhino sporidium seeheri: Preliminary report. J LaryngolOtol 1989;103:178-180. 18 Symmers WStC: Deep-seated fungal infections currently seen in the histopathologic service of a medical school laboratory in Britain. Am J Clin Pathol 1966;46:514-537.
Ravi/Mallikarjuna/Chaturvedi
P enile R h in o sp o rid io sis
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R eferences
Departments of Genitourinary Surgery and Pathology, Cancer Institute (WIA), Adyar, Madras, India
Rhinosporidiosis Mimicking Penile Malignancy
K e y w o rd s
A b stra ct
Penis Penile neoplasms Rhinosporidiosis
Rhinosporidiosis is a chronic infection which is endemic in India and Sri Lan ka. Penile involvement in rhinosporidiosis is rare and we report one such case. The pathology, treatment and possible etiology of the condition are re viewed.
Rhinosporidiosis is a chronic granulomatous disease caused by Rhinosporidium seeberi, an endosporulating organism that is usually classified among fungi [1], The disease is endemic in South India and Sri Lanka, 9 out of every 10 cases reported in the world literature being from this region [2, 3], The lesion usually presents as a friable, pedunculated or sessile, polypoid mass involving mainly the nasal mu cosa in 70% of the cases [2], The less common sites of involvement include the nasopharynx, maxillary antrum, conjuctiva, tracheobronchial tree, larynx, skin, vagina, vulva and rectum [4, 5], Rarely, involvement of bone has been described [6], The lesion is rarely seen in western countries especially if located outside the nasal cavity. Penile involvement in rhinosporidiosis is a rare occur rence and herein we report one such case.
Case Report A 55-year-old male presented to the urology services with a 10year history of painless, polypoid lesions over the glans and external urethral meatus. The lesions were first seen over the external meatus but gradually spread to involve the glans penis. The lesions occasion ally bled and fell off but never regressed completely. The lesions were excised elsewhere 6 years ago but recurred and he was referred to our
Received: November 19. 1991 Accepted after revision: March 10. 1992
centre as a case of penile malignancy. The patient used to bathe regu larly in a temple pond frequented by many people. On examination, multiple polypoid and vascular lesions measur ing about 2.0 cm were seen arising from the urethra over the external meatus and fossa navicularis. In addition, the entire surface of glans was studded with smaller lesions (fig. 1). A biopsy of the lesion was done. Histopathology showed edematous fibrovascular tissue lined by hyperplastic squamous epithelium. The subcpithelial tissue showed a large number of sporangia measuring 10-200 pm and con taining hundreds of endospores (fig. 2). Some of the sporangia had ruptured and endospores were seen in the surrounding tissue. A diag nosis of rhinosporidiosis was made. In view of the extensive involve ment of the glans, a partial amputation of penis was carried out. The patient is doing well 6 months postoperatively without any recur rence.
D iscussion
Rhinosporidiosis of the penis is a rare lesion. It was first reported by Ingram [7] in 1910 and since then about 37 cases have been reported in the English literature [810]. The disease manifests as urethral involvement usually restricted to the fossa navicularis and external ure thral meatus. The lesions are seen as friable, vascular, polypoid or sessile masses arising from the terminal ure thra. Involvement of the glans is unusual and extensive involvement can mimic a penile neoplasm [7],
Dr. R. Ravi. Assistant Professor Department of Genitourinary Surgery Division of Surgical Oncology Cancer Institute (WIA) Adyar. Madras 600 020 (India)
© 1992 S. Kargcr AG. Basel 0042-1138/92/0494-0224 $2.75/0
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/31/2018 12:16:09 PM
R. Ravia V.S. Mallikarjunab H.K. Chaturvedia
Fig. 2. Mature sporangium of R. seeberi containing endosporcs seen in the stroma of the penile lesion along with trophocytes in var ious stages of development. HE X 250.
The disease is caused by Rhinosporidium seeberi, an organism first described and identified as a protozoan by Seeber [ 11 ] in 1900. Ashworth [ 12] in 1923 suggested it to be a fungus and it is classified under phycomycetes although its fungal nature has been disputed [13]. Al though earlier efforts to culture the organisms [8, 14, 15] or transmit them to laboratory animals [2, 16] were unsuccessful, recently the organisms have been cultured by Krishnamoorthy et al. [17]. These authors found posi tive cultures in 92% (23 of 25) of their cases and demon strated it to be a fungus because of the presence of conidium. In its life cycle, the early trophocyte is about the size of a red blood cell, covered by chitin and lies freely in the con nective tissue. It gradually increases in size and the wall thickens. At the size of 45 pm diameter, the nucleus begins to divide. By the 14th division, the trophocyte becomes 140 pm in diameter and contains 16,000 young spores, each measuring 3 pm in diameter. Maturation of the spores occurs with the formation of mucoid envelope with chitinoid film, enlargement to 7-9 pm and the appearance of refringent spherules, 10-16 in number. The mature spo rangium is about 200-250 pm in diameter and it ruptures to release the spores through the germinal pore. Although the exact mode of transmission has not been clearly established, it is likely that contaminated water
and soil is the source of infection. This is strengthened by the observation that the disease is more common in males, particularly from rural areas, who have a habit of swimming in stagnant water. The disease is also found in a variety of animals like horses, mules, cattle and dogs and could possibly be transmitted to humans by contami nated water. Samaddar and Sen [5] found that more than 90% of their 166 patients with nasal rhinosporidiosis had the habit of bathing in ponds frequented by animals. The question of sexual transmission has been raised by the description by Symmers [ 18] of vulval rhinosporidiosis in a female and penile urethral rhinosporidiosis in a male, both of whom were sexual partners. However, similar evi dence of sexual transmission or sexual aberrations in patients with penile rhinosporidiosis has not been found by others [10]. Medical treatment in the form of trivalent and pentavalent antimony compounds has been tried and found to be of little value. Surgical excision of the lesion(s) is the treatment of choice. Partial amputation may be necessary if there is diffuse involvement of the glans as well. Recur rences are seen in up to 25% of patients with urethral rhi nosporidiosis [10], However, its incidence can be de creased considerably by the use of a diathermy knife for excision and electrocauterisation of the base of the le sion.
225
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Fig. 1. Multiple polypoid lesions are seen arising from the exter nal urethral meatus and fossa navicularis. The tip of the glans is swollen and covered with minute lesions. The probe is in the ure thra.
1 Friedman I, Osborn DA: Pathology of Granu lomas and Neoplasms of the Nose and Para nasal Sinuses. Edinburgh. Churchill Living stone, 1982, pp 1-64. 2 Karunaratne WAE: Rhinosporidiosis in Man. London, Althone Press, 1964, p 21. 3 Kameswaran S: Surgery in rhinosporidiosis. Experience with 293 cases. Int J Surg 1966;46: 602-605. 4 Mohapatra LN: Rhinosporidiosis; in Baker RD (ed): Human Infection with Fungi, Actinomy cètes and Algae. Berlin, Springer, 1971, chap 14, pp 676-683. 5 Samaddar RR, Sen MK: Rhinosporidiosis in Bankura. Indian J Pathol Microbiol 1990:33: 129-136. 6 Aravindan KP. Viswanathan MK, Jose L: Rhi nosporidiosis of bone. Case report. Indian J Pathol Microbiol 1989;32:312-313. 7 Ingram AC: Rhinosporidium kinealyi in un usual situations. Lancet 1910;ii:726.
226
8 Dhayagude RC: Unusual rhinosporidial infection in man. Indian Med Gazette 1941 ;76:513— 515. 9 Palaniswamy R, Bhandari M: Rhinosporidio sis of male terminal urethra. J Urol 1983:129: 598-599. 10 Sasidharan K, Subramanian P, Moni VN, Aravindan KP, Chally R: Urethral rhinosporidiosis. Analysis of 27 cases. Br J Urol 1987; 59:66-69. 11 Seeber GR: Un nueva esporonario parasite del hombre. Dos casos encontractos an polipos nasales. Jesis, Univ Mac de Buenos Aires, 1900, pp 1-620. 12 Ashworth JH: On Rhinosporidium seeheri (Wernicke, 1903), with special reference to its sporulation and affinities. Trans R Soc Edin burgh 1923;53:301-342. 13 Vanbreuseghem R, Devroy CH, Takashio M: Practical Guide to Medical and Veterinary' My cology. New York, Masson, 1978, p 227.
14 Rajan RV, Viswanathan GG, Rao AR, Rangiah PN, Anguli VC: Rhinosporidiosis: A study with report of a fatal case of systemic dissemi nation. Ind J Surg 1955;17:269-298. 15 Satyanarayana C: Rhinosporidiosis with a rec ord of 255 cases. Acta Otolaryngol 1960;51: 348-366. 16 Reddy DG, Lakshminarayana CS: Investiga tion into transmission, growth and serology in rhinosporidiosis. Indian J Med Res 1962;50: 363-369. 17 Krishnamoorthy S, Seerdharan VP. Koshy P. Kumar S, Anilkumari CK: Culture of Rhino sporidium seeheri: Preliminary report. J LaryngolOtol 1989;103:178-180. 18 Symmers WStC: Deep-seated fungal infections currently seen in the histopathologic service of a medical school laboratory in Britain. Am J Clin Pathol 1966;46:514-537.
Ravi/Mallikarjuna/Chaturvedi
P enile R h in o sp o rid io sis
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/31/2018 12:16:09 PM
R eferences
