Primary Penile Squamous Cell Cancer-Related Malignant Priapism in a Cystectomized Patient: A Case Report

Priapism is one of the most common urological emergencies and is characterized by a prolonged and painful erectile condition that is not associated with sexual stimulation or sexual desire. Priapism associated with a neoplasm is a rare condition and is usually caused by bodily metastases from other pelvic malignancies. Primary malignant priapism associated with penile malignancy is extremely rare. In this reported case, an 82-year-old man had priapism. Penile Doppler ultrasound and pelvic magnetic resonance were compatible with ischemic priapism and body mass. After that, the patient underwent total penectomy and bilateral superficial inguinal lymphadenectomy. The pathology report was consistent with primary penile squamous cell carcinoma (SCC), so the patient underwent adjuvant radiotherapy. However, he developed multiple metastases and was able to survive for about six months. The patient underwent radical cystectomy (RC) and urethrectomy 19 and 2 years ago for urothelial carcinoma, respectively. To our knowledge, this is the second case of malignant priapism due to primary penile SCC and represents one of the longest periods of urethral recurrence after RC. When a patient has malignant priapism, a primary penile malignancy should be considered in the differential diagnosis, even if the patient has a history of pelvic malignancy.

introduction

Priapism is a prolonged (more than four hours) and unwanted erection without sexual stimulation or the appearance of sexual desire [1]. May be secondary to hematological diseases such as sickle cell disease, infections, metabolic diseases, drugs and neoplasms [2,3]. Priapism due to a neoplasm is called malignant priapism, which is a rare condition that occurs with body metastases of other malignancies in the pelvic area. [4]. Priapism due to primary penile malignancies is extremely rare [5].

In this report, we present the case of a patient who underwent total penectomy and bilateral inguinal lymphadenectomy for malignant priapism associated with primary penile squamous cell carcinoma (SCC), as well as radical cystectomy (RC) and urethrectomy 19 and 2 years ago for urothelial carcinoma (UC). , respectively.

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Case presentation

An 82-year-old patient came to the clinic with an involuntary and painful erection of the penis lasting 48 hours. The patient’s clinical history indicated that he underwent RC and orthotopic neobladder 19 years ago for invasive UC of the bladder. No tumor was observed in the pathology of the final cystectomy (T0); therefore, no adjuvant therapy was given. He underwent total urethrectomy, non-urinary bladder resection, and ileal conduit 17 years after RC surgery for recurrent midurethral UC. The urethral pathology report was consistent with high-grade urothelial carcinoma with subepithelial invasion (high-grade pT1). Adjuvant chemotherapy based on cisplatin was applied. During the patient’s examination, a cold, erect penis with an ulcerated lesion of 1 cm in diameter was observed on the head of the penis. Bedside body irrigation and aspiration were performed, and a cavernous blood gas sample was sent for differential diagnosis. Hypoxia, hypercarbia and acidosis were observed on cavernosal blood gas examination (pH:7.01, pO2: 27 mmHg, pCO2: 78 mmHg), which was compatible with ischemia. The blood count was normal. Detumescence could not be achieved, and to evaluate additional pathologies, the patient underwent an emergent duplex Doppler ultrasound of the penis, which showed reduced blood flow and a mass in the proximal part of the penis. An MRI of the pelvis was then performed and a mass was observed that was completely destructive to the corpora cavernosa and had irregularly rounded borders and necrotic areas; the border with the pubis was unclear (Fig 1).

Pelvis-magnetic-resonance-imaging-patient.

Later, the patient underwent total penectomy and bilateral inguinal lymphadenectomy. The report from the penectomy specimen suggested that the tumor was compatible with moderately differentiated SCC. Extensive tumor sampling did not reveal UC in situ or SCC in situ. The bilateral corpus cavernosum is completely infiltrated by the tumor, with SCC morphology starting from the surgical margin and damaging all structures of the penile body (Figure 2a). For a possible contribution to the differential diagnosis, p16 immunohistochemistry was performed, but the tumor was negative. Since p16 is used as a surrogate marker for human papillomavirus (HPV)-induced carcinomas, its positivity would support HPV-driven primary SCC in this case as well. However, since it was negative, it did not help the pathologist to make a final diagnosis (Fig 2b). No signs of lymph node metastasis were observed (pT3N0). The tumor was positive at the proximal surgical margin. After that, the patient underwent adjuvant pelvic radiotherapy; however, due to the worsening of his general condition, he could not continue radiation therapy and receive chemotherapy; he later developed multiple metastases and was only able to survive for about six months.

Discussion

Malignant priapism is a rare clinical condition caused by primary or secondary penile malignancies. It was first described by Peacock in 1938 as a persistent and painful erection resulting from invasion of cavernous sinuses and efferent veins. [4]. Malignant priapism is caused by secondary penile tumors, not primary tumors. The most common sources of body metastases are bladder, prostate, and recto-sigmoid tumors, in that order. [6]. The size, location, presence of priapism, and prognosis of the underlying tumor affect the treatment of penile metastases [7]. Local excision, partial or complete penectomy, radiation, chemotherapy and conservative approach are some of the treatment options [7,8].

Cases of priapism due to primary penile malignancies are extremely rare. Priapism due to primary penile malignancies has been reported in six cases in the literature [9-11]. While four cases had primary penile lymphoma, one case had penile hemangioendothelioma, and only one case had primary penile SCC. In a case of primary penile SCC, a 37-year-old patient had priapism, and a penile biopsy revealed poorly differentiated SCC. Since the general condition of the patient was bad, external radiotherapy was applied; however, the patient’s condition worsened over the next two months, and he succumbed to his illness [10]. There are no established protocols for the treatment of malignant priapism caused by penile squamous cell carcinoma. Since malignant priapism due to primary penile malignancies is a locoregional disease as opposed to a secondary priapism-inducing one, multimodal therapy may be curative. In the case presented in this report, we performed total penectomy as well as bilateral inguinal lymphadenectomy in this 80-year-old patient who had severe pain due to malignant ischemic priapism and whose penis had no urinary function due to previous RC and urethrectomy operations, followed by pelvic radiotherapy. However, this treatment was not sufficient to control the cancer, and the patient developed multi-metastases.

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Conclusions

In conclusion, we report a patient who presented with primary malignant priapism associated with penile SCC and a history of bladder and urethral cancer. Despite aggressive local treatment, the patient could only survive for six months. Although it is the most common primary penile malignancy, to our knowledge, priapism due to primary penile SCC has only been reported once in the literature. Although metastatic disease is usually thought to be the cause, when a patient with a history of pelvic malignancy presents with malignant priapism, it should be kept in mind that a primary penile malignancy may also be the etiology. As the data on patients with malignant priapism associated with primary penile cancer increases, therapeutic guidelines can be created for the treatment of this condition.



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