0022-5347/95/1533-105 1$03.00/0 Tm JOURNAL OF UROLOGY Copyright 0 1995 by AMZRIC&V UROLOGICN. ASSOCIATIOS, INC.
Vol. 153,1051-1052,March 1995 Printed in U.S.A.
PRIMARY PENILE LYMPHOMA PRESENTING AS A PENILE ULCER CYNTHIA A. FAIRFAX, CHARLES J. HAMMER, 111, BRUCE W. DANA, JON M. HANIFIN AND
JOHN M. BARRY From the Divisions of Urology, and Hematology and Oncology, Department of Dermatology, The Oregon Health Sciences Uniuersity, Portland, Oregon
Primary penile lymphoma is rare. The clinical manifestations can be quite subtle, which may lead to misdiagnosis. Treatments have included radical surgery, chemotherapy and radiation. We report on a n 18-year-old man with primary penile lymphoma whose lesion caused prolonged diagnostic uncertainty. He was treated with chemotherapy alone, and h e has had no tumor recurrence for 27 months.
KEY WORDS:lymphoma, penis, drug therapy, ulcer Soft tissue tumors of the penis occur in 1to 2 per 100,000 cases per year in the United States.‘ Primary penile lymphoma is extremely rare, and we are aware of only 9 previously reported The longest disease-free survival reported is 6 years after treatment with total penectomy and systemic chemotherapy.* Marks et a1 reported disease remission and minimal disfigurement with chemotherapy alone.3 The latter approach was used in our case, and it resulted in prompt healing of the ulcerated lesion without recurrence at 27-month followup. CASE REPORT
An 18-year-oldcircumcised man presented with a 7-month
history of a painless penile ulcer. He denied self-mutilation. The lesion began as a white pustule on the dorsum of the penis proximal t o the corona, and it gradually enlarged and became a 2.0 X 1.5 cm. ulcer. Physical examination was otherwise normal. The patient was treated for presumed venereal disease with several courses of antibiotics and anFIG. 1. Penile ulcer subsequent to second biopsy and before tifungal agents as well as local wound care without improvement. Tests for chlamydia, gonorrhea, syphilis,herpes sim- chemotherapy. plex virus, human immune deficiency virus and fungi were negative. Three months after the lesion appeared, it was debrided and biopsied with findings of “marked acute and chronic inflammation and granulation tissue.” Due to an unstable social situation and the negative diagnostic evaluation of the penile lesion, a factitious injury was suspected. Psychiatric evaluation revealed no evidence of psychosis. After 2 additional months of evaluation and therapy without improvement the patient was admitted to our hospital. The lesion now measured 3 X 5 cm. and occupied the entire dorsum and half the circumference of the penis (fig. 1). There was no evidence of infection. The patient also had daily temperature elevations to 38.9C. Extensive dkbridement and biopsy revealed diffise large cell immunoblastic lymphoma extending into the corpora cavemosa (fig. 2). Metastatic evaluation included a normal bone marrow aspirate and chest radiograph. Computerized tomography of the abdomen and pelvis showed a small low density lesion in the anterior right lobe of the liver but needle biopsy revealed no evidence of FIG. 2. Penile lymphoma with large cells, abundant cyto l a m lymphoma. The tumor was stage IE (single extra lymphatic and large convoluted or multilobulated nuclei. Multinucleatef cells site according to the Ann Arbor staging classificationg)with were present and mitotic figures conspicuous. Reduced from x400. B symptoms (fever). Therapy consisted of 8 courses of cyclophosphamide, doxo- had regular followup evaluations and remains free of recurrubicin, vincristine and prednisone. During the 7-month rence 27 months after diagnosis. course of treatment the ulcer gradually healed but with significant scar (fig. 3). However, the patient reported normal DISCUSSION erectile function and did not desire cosmetic surgery. He has The presentation of penile lymphoma varies. Indurated plaques, nodules, ulcers and diffise penile swelling have Accepted for publication July 8, 1994. 1051
PRIMARY PENILE LYMPHOMA PRESENTING AS PENILE ULCER
Reported cases of penile lymphoma Reference
Follawup Died 1 mo. aRer diagnosis Not stated Lost to followup
Oomura et a12
Surgery and chemotherapy
Cough' Dehner and Smith'
Reticulum cell sarcoma Small cell lymphosarcoma Reticulum cell sarcoma Lymphocytic lymphoma Follicular nododgkin's lymphoma Lymphoma, possibly T-lineage B-lineage large cell lymphoma Diffise large cell lymphoma
Surgery Not stated Radiation Surgery and chemotherapy Radiation Radiation Surgery and chemotherapy Chemotherapy
Gonzales-Campora et al' Stewart et ale
Yu et als Marks et d3
56 76 60 54 60 65
Not stated 3 yrs. 1 .w. 6 yrs. Not stated
Not stated None Not stated None None None Not stated ____
ment or loss of erectile function. Y u et al suggested that penile lymphoma is a manifestation of occult nodal disease.' Metastatic malignant tumors of the penis are rare with less than 200 cases reported in the literature, and less than 1%of these tumors are of lymphoid origin." Only 1 case in our review of primary penile lymphoma documented inguinal node involvement.2 However, this possibility gives further reason to treat with systemic chemotherapy. Penile malignancies may be easily overlooked in young, healthy individuals. Our case suggests the need for repeated and thorough biopsy for persistent genital lesions that do not have an identifiable infectious or traumatic etiology. Isolated penile lymphoma can be especially difficult to diagnose. However, good prognosis and reasonable preservation of penile function are possible with appropriate treatment. Dr. Makoto Nakamura provided translation assistance. REFERENCES
1. Schellhammer, P. F., Jordan, G. H. and Schlossherg, S. M.: FIG. 3. Healed ulcer 4 months aRer initiating chemotherapy Tumors of the penis. In: Campbell's Urology. 6th ed. Edited by P. C. Walsh, A. B. Retik, T. A. Stamey and E. D. Vaughn. Philadelphia: W. B. Saunders Co., vol. 2. chapt. 31. pp. 12641291,1992. been r e p ~ r t e d . ~It. ~can . ~ mimic an infectious process, carci2. Oomura, J., Ookita, K., Takenaka, M. and Yamada, S.: Primary surnoma or injury. Treatment has included ~hemotherapy,~ gery: radiotherapy's6 and surgery plus c h e m ~ t h e r a p y ~ . ~ . ~ reticulosarcoma o f the penis: report of a caHe. Hinyokika Kiyo, 8 536,1962. (see table). Reported 2-year disease-free survival after che3. Marks, D.,Crosthwaite. A,, Varigos. G., Ellis. D. and Morstyn, ' motherapy has been as high as 83%for extranodal large cell G.: Therapy of primary diffuse large cell lymphoma of the lymphomas (stage lEh3 penis with preservation of function. J. Urol., 139 1057,1988. The difliculties in the diagnosis of this rare malignancy are 4. Gough, J.: Primary reticulum cell sarcoma of the penis. Brit. J. , demonstrated in our case. The puzzling presentation of a Urol., 42: 336, 1970. 5. Dehner, L. P. and Smith, B. H.: SoR tissue tumors of the penis. penile ulcer, combined with the nondiagnostic initial biopsy, A clinicopathologic study of 38 patients. Cancer, 25: 1431, led to a strong suspicion of self-mutilation. The young man, 1970. although under profound stress, did not appear to have psy6. Stewart, A. L., Grieve, R. J. and Banerjee, S. S.: Primary lymchiatric disease. While most genital self-mutilations are asphoma of the penis. Eur. J. Surg. Oncol., 11: 179,1985. sociated with psychosis, there have been reports of mutila7. Gonzalez-Campora, R.,Nogales, F. F., Jr., Lerma, E., Navarro, tion for erotic purposes or as part of a tribal ritual, and A. and Matilla, A.: Lymphoma of the penis. J. Urol., 126 270, nonpsychotic genital mutilation may not be as uncommon as 1981. originally thought." 8. Yu, G. S.M., Nseyo, U. 0. and Carson, J . W.: Primary penile Persistence in evaluation and re-biopsy of this lesion relymphoma in a patient with Peyronie's disease. J. Urol., 142 1076,1989. sulted in the correct diagnosis and appropriate treatment. 9. Glick, J . H.: Hodgkin's disease. In: Cecil Textbook of Medicine, Whether delay in diagnosis will affect long range outcome is 19th ed. Edited by J. B. Wyngaarden, L. H. Smith, Jr. and unknown. Favorable outcome with complete remission and J. C. Bennett. Philadelphia: W. B. Saunders Co., chapt. 148, preservation of penile function with chemotherapy alone has pp. 955-963, 1992. chemotherapy the lesion in our 10. Greilsheimer, been d ~ c u m e n t e d After .~ H.and Groves, J. E.: Male genital self-mutilation. patient healed without the need for skin grafting. Other Arch. Gen. Psychiat., 3 6 441,1979. successful therapeutic modalities for stage IE large cell lymph- 11. Osther, P. J. and Untoft, E.: Metastasis to the penis: case , omas have been mentioned previously. Each of these other reports and review of the literature. Int. Urol. Nephrol., 23 treatments can result in significant morbidity with disfigure161,1991. ~