Case Report

Urologia

Received: April 2, 2014 Accepted after revision: April 23, 2014 Published online: August 13, 2014

Urol Int 2016;96:244–246 DOI: 10.1159/000363113

Internationalis

‘Prostate Abscess’ as the Initial Manifestation of Granulomatosis with Polyangiitis (Wegener’s Granulomatosis) Sotirios Tsiodras a Garyfalia Poulakou a Konstantinos Leventakos a Helen Panopoulou b Antonia Elezoglou a Kassiani Manoloudaki d Michail Chrisofos c George Petrikkos a Ioannis G. Panayiotides b   

 

 

 

 

 

 

 

 

a 4th Department of Internal Medicine, b 2nd Department of Pathology and c 2nd Department of Urology, University of Athens Medical School, ‘Attikon’ University Hospital, Athens, and d Department of Pathology, ‘Tzaneion’ General Hospital, Piraeus, Greece  

 

 

 

Abstract Prostatic involvement in granulomatosis with polyangiitis (GWP), formerly known as Wegener’s granulomatosis, is rare, mostly arising in the context of systemic involvement. Prostatic involvement as the first manifestation of this systemic disease is exceptionally rare. We hereby present the case of a 41-year-old male patient who underwent transurethral prostate resection for what was initially diagnosed as suppurative, focally necrotizing prostatitis. Prolonged postoperative fever that did not respond to various treatments, as well as the subsequent appearance of a left pleural effusion, a left upper pulmonary lobe lesion and cutaneous nodules, led to a reevaluation of histological slides which, along with the determination of serum c-ANCA/anti-PR3 antibody levels, established the diagnosis of GWP. Physicians, and especially urologists and infectious diseases specialists, should

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be aware of this rare association and consider GWP in the event of nonresolving prostatitis, especially when characteristic symptoms from other systems appear. © 2014 S. Karger AG, Basel

Introduction

Initially described in 1939, Wegener’s granulomatosis is currently included in the group of antineutrophil cytoplasmic antibodies (ANCA)-associated vasculitides, along with Churg-Strauss syndrome and microscopic polyangiitis [1]. The term ‘granulomatosis with polyangiitis’ (GWP) was recently proposed as an alternative term for the disease due to Friedrich Wegener’s membership of the Nazi Party (NSDAP) during World War II [2] but has not yet achieved widespread use. The classic diagnostic triad of GWP includes the presence of the following: (1) acute necrotizing granulomatous inflammation of the upper respiratory tract and the lungs, (2) focal necrotizing arteritis of small-to-mediumsized vessels and (3) renal involvement, most often in the Ioannis G. Panayiotides, MD, PhD 2nd Department of Pathology, University of Athens Medical School ‘Attikon’ University Hospital, 1 Rimini Street, Chaidari GR–12462 Athens (Greece) E-Mail ioagpan @ gmail.com

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Key Words Prostate abscess · Granuloma · Prostate · Prostatitis · Granulomatosis with polyangiitis · Wegener’s granulomatosis

Case Report

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form of focal segmental glomerulonephritis. It is usually associated with the detection of circulating ANCA (c-ANCA) against serine protease-3 (PR3). Its etiology and pathogenesis has not yet been fully elucidated, although various putative antigens have recently been invoked in the context of a local autoimmune reaction within the granuloma [3]. The prevalence of GWP amounts to 3:100,000, with no gender differences. Lower urogenital involvement is rare and mostly concerns male patients [4], with the penis, testes, seminal vesicles and bladder being involved in decreasing order of frequency [5]. Prostatic involvement is rare, mostly in the context of systemic involvement. Very rarely, prostatic involvement has been reported as the first manifestation of the disease. We hereby present such a case, initially misdiagnosed as suppurative prostatitis.

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Fig. 1. Histopathological findings from the prostate biopsy are depicted. a Prostate chips. HE. ×2. b An area of geographic necrosis. HE. ×4. c Multinucleate giant cells (arrow) and necrosis. HE. ×10. d Vasculitis. HE. ×20.

A 41-year-old male was admitted to the department of urology of a general hospital due to dysuria and fever up to 38.5 ° C. Urine analysis disclosed pyuria and hematuria, without the presence of glomerular RBCs in the urine specimen. Transrectal prostate ultrasound and abdominal CT disclosed findings consistent with the presence of a prostatic abscess surrounded by diffuse edema. Urethral discharge and urine cultures yielded Staphylococcus epidermidis and Enterococcus faecalis. Despite being treated with various antibiotic regimens, his symptoms did not improve and there was no radiological improvement of the abscess; he therefore underwent a transurethral resection of the prostate. A histological diagnosis of acute, suppurative, focally necrotizing prostatitis was rendered. Persisting postoperative fever did not respond to various antibiotic combinations; moreover, the patient developed a left pleural effusion. Besides the effusion, thoracic CT disclosed a nodule located at the left upper pulmonary lobe. Pleural fluid cultures were sterile, whereas a Ziehl-Neelsen stain and cultures showed no acid-fast bacteria. A bronchoscopically obtained biopsy showed acute inflammatory reaction with no vasculitis or granulomas. The patient was transferred to the department of internal medicine of a university hospital. Upon admission, he had a fever of 39.1 ° C, tachycardia, four cutaneous red, tender nodules (located on both legs and left arm) and a red left eye; he was also mildly confused. Cranial CT and ophthalmic examination were unremarkable. Laboratory examinations disclosed high CRP levels and a 1: 50 c-ANCA titer concerning anti-PR3 antibodies; urine sediment contained leukocytes, whereas BUN, creatinine and electrolyte testing was within normal limits. Histological examination of a cutaneous nodule showed a nonspecific dermal acute inflammation, with no granulomas or vasculitis. Nevertheless, a review of slides from the transurethral resection of the prostate disclosed irregular (socalled ‘geographic pattern’) areas of ‘dirty’ necrosis (i.e. containing karyorrhectic debris and polymorphonuclear leukocytes), foci of vasculitis and (upon multiple, consecutive sections) a single, illformed granuloma, thus confirming the clinical suspicion of GWP (fig. 1). Periodic acid-Schiff, Grocott and Ziehl-Neelsen stains were

Prostatic involvement in GWP is rare, amounting to 2.3–5% of cases [6, 7]. It mostly arises in the context of systemic involvement [4]. Very rarely, prostatic involvement has been reported as the first manifestation of the disease [8]. Histology usually reveals necrotic granulomas [7]. Nevertheless, GWP is a very rare cause of granulomatous prostatitis, amounting to just 2% in a previously published series of 200 cases [9]. Other causes of granulomatous prostatitis include infections (such as tuberculosis or mycoses), previous intravesical BCG treatment, previous transurethral resection, sarcoidosis that has being vanishingly rare, or ‘idiopathic’ forms of the disease where no cause was identified [7, 10]. Morphology of the granulomas (e.g. palisading type in postresection cases), as well as ancillary techniques (histochemical stains, PCR for fungi or mycobacteria) together with serological testing for anti-neutrophilic antibodies, assists in the differential diagnosis of this rare entity. Other autoimmune diseases seldom manifest with initial or even exclusive involvement of the prostate, such as giant cell

Granulomatosis with Polyangiitis (Wegener’s Granulomatosis) in Prostate

Urol Int 2016;96:244–246 DOI: 10.1159/000363113

 

 

 

negative for fungi or acid-fast bacteria. The patient was started on mycophenolate mofetil and corticosteroids, with a rapid improvement of his symptoms. He was discharged under follow-up by the rheumatology clinic on an outpatient basis and has been doing well with no recurrence of his prostate symptoms.

Discussion

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arteritis, polyarteritis nodosa, microscopic polyangiitis and Churg-Strauss syndrome [7]. The management of this rare entity is etiological and depends upon the establishment of the correct diagnosis. Recent studies describe the use of molecular techniques to identify rare pathogens in patients with prostate disease [11] and where percutaneous drainage of the abscess could have been attempted [12]. However, only after prostate resection were we able to diagnose our patient’s condition. In conclusion, we describe a rare case where prostatic involvement was not only the first manifestation of GWP but was also managed for a significant period as a prostatic abscess leading to prostate resection. GWP prostate involvement is a rare entity, arising mostly in the context

of systemic involvement, as eventually occurred in the case presented herein. Physicians, and especially urologists and infectious diseases specialists, should be aware of this rare association and consider GWP in the event of nonresolving prostatitis, especially when characteristic symptoms from other systems appear.

Disclosure Statement None of the contributing authors have any conflict of interest, including specific financial interests or relationships and affiliations relevant to the subject matter or materials discussed in this paper.

References

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5 Middleton G, Karp D, Lee E, Cush J: Wegener’s granulomatosis presenting as lower back pain with prostatitis and ureteral obstruction. J Rheumatol 1994;21:566–569. 6 Stillwell TJ, DeRemee RA, McDonald TJ, et al: Prostatic involvement in Wegener’s granulomatosis. J Urol 1987;138:1251–1253. 7 Petersen RO, Sesterhenn IA, Davis CJ: Prostate; in Urologic Pathology, ed 3. Philadelphia, Lippincott Williams & Wilkins, 2009, pp 451–560. 8 Gunnarsson R, Omdal R, Kjellevold KH, Ellingsen CL: Wegener’s granulomatosis of the prostate gland. Rheumatol Int 2004; 24: 120– 122.

9 Stillwell TJ, Engen DF, Farrow GM: The clinical spectrum of granulomatous prostatitis: a report of 200 cases. J Urol 1987;138:320–323. 10 Maurice MJ, Zhu H: Sarcoidosis of the prostate. J Urol 2013;190:711–712. 11 Xiao J, Ren L, Ly H, Ding Q, Lou S, Zhang W, Dong Z: Atypical microorganisms in expressed prostatic secretion from patients with chronic prostatitis/chronic pelvic pain syndrome: microbiological results from a casecontrol study. Urol Int 2013;91:410–416. 12 Arrabal-Polo MA, Jimenez-Pacheco A, Arrabal-Martin M: Percutaneous drainage of prostatic abscess: case report and literature review. Urol Int 2012;88:118–120.

Tsiodras  et al.  

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1 Gadola SD, Gross WL: The renaissance of granulomatous inflammation in AAV. Nat Rev Rheumatol 2012;8:74–76. 2 Falk RJ, Gross WL, Guillevin L, et al: Granulomatosis with polyangiitis (Wegener’s): an alternative name for Wegener’s granulomatosis. Arthritis Rheum 2011;63:863–864. 3 Thurner L, Muller A, Cerutti M, et al: Wegener’s granuloma harbors B lymphocytes with specificities against a proinflammatory transmembrane protein and a tetraspanin. J Autoimmun 2011;36:87–90. 4 Dufour JF, Le Gallou T, Cordier JF, et al: Urogenital manifestations in Wegener’s granulomatosis. A study of 11 cases and review of the literature. Medicine 2012;91:67–74.

'Prostate Abscess' as the Initial Manifestation of Granulomatosis with Polyangiitis (Wegener's Granulomatosis).

Prostatic involvement in granulomatosis with polyangiitis (GWP), formerly known as Wegener's granulomatosis, is rare, mostly arising in the context of...
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