Spontaneous renal hemorrhage secondary to choriocarcinoma in a man with congenital hypospadias and cryptorchidism: a case report and literature review

Abstract

BackgroundChoriocarcinoma is a rare malignant germ-cell tumour, most commonly found in adult women. It infrequently presents as spontaneous renal haemorrhage (SRH). Genital malformation and SRH secondary to choriocarcinoma has previously been only reported in females. We present what we believe to be the first case of a male patient with genital malformation (hypospadias and cryptorchidism) and SRH at presentation of choriocarcinoma.Case presentationA 25-year-old man presented to the department with intense pain in the right flank region and lower back. Initial investigations showed spontaneous renal haemorrhage, for which an emergency partial nephrectomy was performed. Clinical, radiological, and pathological investigations suggested a diagnosis of testicular choriocarcinoma with metastases to the right kidney, both lungs, and brain. Initial treatment was with a chemotherapy regimen of cisplatin, etoposide and bleomycin and whole brain radiotherapy; however, 6 months after diagnosis the patient developed liver metastasis, after which time the BEP protocol was switched to ITP with oral apatinib. Despite best efforts, the liver and lung metastasis continued to grow and a decision was made to discontinue active treatment and provide only palliative care until the patient passed away.ConclusionChoriocarcinoma is a difficult cancer to diagnose pre-operatively. In male patients with early metastasis, prognosis may be much poorer than in the commoner gestational choriocarcinoma. A multidisciplinary with comprehensive post-surgical intervention is of great importance in the treatment of these patients.

Case Report: 18F-FDG PET/CT and Laparoscopic Nephron Sparing Surgery in the Management of Bleeding From Renal Metastases of Choriocarcinoma

“…In a similar case, Li et al. ( 9 ) used the open surgical approach and partial nephrectomy to control the bleeding. In the cases of Vijay et al.…”

Nontraumatic spontaneous bilateral renal hemorrhages (Wünderlich syndrome) with chronic infection of Chlamydia pneumoniae: A case report

“…Spontaneous nontraumatic renal hemorrhage was rst documented in the literature by Bonet in 1700 as a rare disease, although it was again reported as Wünderlich syndrome by a German doctor in 1856. [1][2][3][4][5][6][7][8][9][10][11][12][13] Spontaneous bilateral renal hemorrhage is extremely rare because it is unlikely to occur in the absence of anticoagulant or antiplatelet intake when it occurred consecutively not simultaneously. 14 In our case, the rst event of the renal hemorrhage occurred when the patient took clopidogrel (oral antiplatelet) daily, and the second event of renal hemorrhage occurred only 15 days after the rst hospital discharge.…”

“…Most cases were caused by vascular proliferative tumors, such as renal cell carcinoma or angiomyolipoma (57-73%) or choriocarcinoma, followed by autoimmune vasculitis including polyarteritis nodosa (PAN) or segmental arterial mediolysis. [1][2][3][4][5][6][7] In particular, spontaneous bilateral renal hemorrhages accounted for only 3% of all case reports on spontaneous renal hemorrhage, and all cases were PAN. 14 Phillips et al reported that BehÇet's disease and segmental arterial mediolysis may cause spontaneous renal hemorrhage.…”

Testicular Choriocarcinoma with Inexplicably High Beta-human Chorionic Gonadotropin and Renal Metastases

“…Li et al reported the case of a 25-year-old man who presented with spontaneous renal hemorrhage, which on further workup was found to be secondary to choriocarcinoma; however, the case was complicated by cryptorchidism and hypospadias. 3 The authors suggested choriocarcinoma was a difficult diagnosis because of its unusual presentation. We report on the second patient to be found with this presentation, who also had widespread disease with metastases in the lungs, mediastinal lymphnodes, and lower pole of the right kidney.…”