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1 October 2014 - Case of the Week #328

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Case of the Week #328

Clinical History:
A 34 year old man had gradually increasing abdominal and left scrotal pain. Scrotal ultrasound demonstrated a left testicular heterogeneous hypoechoic lesion with no significant internal vascularity (figures 1, 2). Radical orchiectomy was performed.

The testis was bivalved to show a hemorrhagic, well-defined, soft, dark red lesion (figure 3).

Scrotal ultrasound:


Gross image:



Micro images:



Additional images:




What is your diagnosis?































Diagnosis:
Testicular necrotizing vasculitis

Discussion:
Microscopic examination of the lesion revealed focal hemorrhage with early organization and small arteries around the area of hemorrhage showing necrotizing vasculitis.

Necrotizing vasculitis is a systemic disease which manifests as inflammation of arteries with fibrinoid necrosis and fibrosis of the vascular wall. It can be associated with a variety of other entities or may be idiopathic (Cardiovascular Pathology 1994;3:197). While necrotizing vasculitis can involve any organ (PubMed Health), isolated testicular involvement is rare and can pose diagnostic difficulties, especially in the absence of clinical and laboratory evidence of systemic involvement (World J Surg Oncol 2011;9:63).

Polyarteritis nodosa (PAN) is the most common form of testicular vasculitis, predominantly seen in the fourth to sixth decades of life. It is often associated with hepatitis B virus and human immunodeficiency virus. Although 60% to 86% of patients with PAN show testicular involvement at autopsy (ScientificWorldJournal 2010;10:1915), testicular symptoms are present in less than 20% of patients (Hum Pathol 1988;19:186). Other vasculitides that may involve the testes include Wegener granulomatosis, Henoch-Schonlein purpura, giant cell arteritis and various autoimmune connective tissue disorders such as systemic lupus erythematosis (J Urol 2006;176:2682).

Testicular vasculitis can present with acute onset of pain and swelling, presumably related to intra-testicular hemorrhage (Ir Med J 2006;99:27). Systemic disease is frequently already present at the time of initial presentation of testicular vasculitis or develops shortly thereafter. Some cases, however, remain isolated to the testes with no evidence of progression to systemic disease (J Clin Pathol 1994;47:1121). It is, therefore, unclear whether isolated testicular vasculitis represents an early site of involvement by systemic disease or a separate entity (World J Surg Oncol 2011;9:63). Only several cases of isolated testicular vasculitis were reported in the literature and a presentation suggestive of a testicular neoplasm is even less common (J Clin Pathol 1994;47:1121). Since testicular biopsy is contraindicated when malignancy is suspected, the diagnosis is usually reported post-orchiectomy in these cases (ScientificWorldJournal 2010;10:1915, Urology 2003;61:1035).

Treatment options are still debated and frequently hinge on individual factors which need to be carefully considered in each patient. Immunosuppressive therapy has been administered to some of the patients. In other cases, careful follow-up to exclude progression to systemic disease was preferred (J Clin Pathol 1994;47:1121). Some authors have even postulated that the excision of the affected organ might be curative, obviating the need for pharmacologic therapy (ScientificWorldJournal 2010;10:1915). Close follow-up, including serology, is recommended since the risk of progression to systemic disease is unknown (J Clin Pathol 1994;47:1121).

Although uncommon, isolated necrotizing testicular vasculitis remains in the differential diagnosis of hypovascular testicular lesions. Long-term follow-up and additional research is needed in order to further elucidate the pathogenesis of isolated organ vasculitis and the subsequent risk of disease progression.


Nat Pernick, M.D., President
and Shivani Thakore, Associate Medical Editor
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