World Journal of Nephrology and Urology, ISSN 1927-1239 print, 1927-1247 online, Open Access
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Case Report

Volume 7, Number 1, March 2018, pages 32-37


Minocycline-Induced Polyarteritis Nodosa Presenting With Testicular Pain: A Case Report and Selected Review of the Literature

Figures

Figure 1.
Figure 1. Left paratesticular tissue with perivascular infiltrates containing of lymphocytes and plasma cells (H&E, 20 × original magnification).
Figure 2.
Figure 2. Left paratesticular tissue with focal necrotizing vasculitis of small and medium sized vessels and perivascular infiltrates containing of lymphocytes and plasma cells (H&E, 40 × original magnification).

Table

Table 1. A Selected Review of the Literature on Testicular Polyarteritis Nodosa [3-18]
 
Study, datePatient age/final DxCCLabs and ultrasoundPathologyTxSx after txSignificance
ANA: anti-nuclear antibody; ANCA: anti-nuclear cytoplasmic antibody; Asymp-: asymptomatic; Avg: average; Bx: biopsy; CRP: C-reactive protein; C/W: consistent with; ESR: erythrocyte sedimentation rate; PAN: polyarteritis nodosa; Sx: symptoms; Tx: treatment; U/S: ultrasound.
Lenert, 2013 [3]21/minocycline-induced systemic PANLeft testicular pain + systemic sxLabs: elevated ESR, CRP, (+) p-ANCA, (+) ANA; U/S: wedge-shaped lesionMedium-size testicular artery with mononuclear cell infiltration and area of testicular necrosisRemoval of minocycline, short course of prednisone and hydroxychloroquineAsymp- and disease free at 2 yearsSystemic vasculitis associated with chronic minocycline use
Gervaise, 2014 [4]28/systemic PANRight testicular painLabs: mildly elevated CRP; U/S: no Doppler flow in right testis with numerous hypoechogenic areas and some areas of normal parenchymaGross: heterogeneous in appearance with alternating areas of ischemia without necrosis and healthy parenchyma c/w acute vasculitisIV prednisolone and cyclophosphamideAsymp-Diagnosis of testicular vasculitis on CT angio: thrombosis of distal testicular artery, no confirmed biopsy
Bing, 2012 [5]46/systemic PANLeft flank pain with gross hematuria after runningLabs: elevated ESR and CRP, anemia, (-) ANA, (-) ANCA; renal U/S: left-sided, upper pole, pelvi-calyceal distension and peri-nephric edemaN/AGlucocorticoid, cyclophosphamide, methotrexateAsym- and disease free at 2 yearsDiagnosis based on presence of aneurysms on left renal angiogram
Toepfer, 2011 [6]55/systemic PANLeft testicular pain, with recurrence on the right 3 weeks after initial evaluation, recurrent systemic ischemic eventsLabs: all (-); U/S: decreased blood flowTesticular specimens: interstial hemorrhage and focal atrophy; Abdominal wall skin bx: thrombotic vasculopathy with leukocytoclastic vasculitisBilateral orchiectomy, methylprednisone + cyclophosphamideRecurrence of ischemic events at 3 weeks and 5 weeks; asymp- after systemic tx at 6 monthsAsynchronous testicular necrosis as initial sign of systemic PAN
Ahmad, 2010 [7]65/systemic PANLeft testicular pain and swelling, developed frank painless hematuria during workupLabs: (+) ANCA, elevated ESR; U/S: mycotic aneurysmal lesions of the testicleN/AGlucocorticoid“Cinically well”Left renal hematoma on CT scan, small aneurysms found in both kidneys on angiography
Meeuwissen, 2008 [8]72, 61, 28/systemic PANTesticular pain preceeding systemic symptoms (1), concurrent systemic sx and testicular pain (2)Labs: elevated ESR, CRP, (-) ANCA, (-) ANA, anemia; U/S: heterogenic, enlarged testisGross: edematous, blue colored testis with multiple necrotic areas; Microscopic: segmental destruction of vessel wall of small and medium-sized arteries by mononuclear inflammatory infiltrate; fibrinoid necrosis and thrombiOrchiectomy, methylprednisolone taper (after onset of systemic symptoms)Asymp- and disease free at 16 months - 80 monthsTesticular involvement as prominent sign of PAN
Kolar, 2007 [9]29/systemic PANRight scrotal pain + systemic sxLabs: elevated ESR, CRP, (-) ANCA; U/S: enlarged and swollen right epididymis with reduced blood flow; reduced testicular artery flowN/AGlucocorticoids + cyclophosphamideN/AImproved testicular artery flow after systemic treatment without need for orchiectomy
Susanto, 2003 [10]74/systemic PANLeft testicular pain + systemic sxLabs: elevated ESR, CRP, anemia, leukocytosis, microscopic hematuria; (-) ANCA; U/S: small right testicular cyst and mild enlargement of left testicle with normal epididymisSkin/muscle bx: vasculitis of medium vessels with mixed cellular infiltrate in intramuscular arterioles and their branchesOral prednisone (1 mg/kg/day) and cyclophosphamide (2 mg/kg/day) × 6 monthsAsymp- at 1 yearFebrile episode of epididymo-orchitis as initial manifestation of PAN
Eilber, 2001 [11]43/systemic PANHematuria, left testicular mass, systemic sxLabs: elevated ESR, (+) ANA; U/S: suspicious heterogeneous intratesticular lesionInflammatory infiltrate and thrombosis in vessel lumina, necrosis of intimal layerOrchiectomy onlyAsymp-PAN presenting with hematuria and testicular lesion
Brimo, 2011 [12]35 (avg) 23-53 (range)/isolated testicular PAN (12), systemic PAN (2)Testicular pain (86%), mass (7%)Labs: all negative except 2 with elevated CRP and ESR, (-) ANCA; 1 with (+) ANA; U/S: hypoechoic mass suspicious for cancerTransmural necrotizing inflammation of small to medium-sized ateries with fibrinoid necrosis and acute inflammationOrchiectomy, systemic treatment: predisone, or prednisone + cytoxan (in half of isolated cases and all of systemic cases)N/ACase report of testicular vasculitis, 14 confirmed to be PAN, half of isolated cases still received systemic treatment
Fraenkel-Rubin, 2002 [13]26/iIsolated testicular PANLeft testicular painLabs: negative ESR, CRP, ANA, ANCA, CBC; U/S: diffuse damage consistent with interstitial processFibrinoid necrosis of medium and small sized arteries with acute transmural inflammation with lymphoplasmacytic and eosinophilic infiltratesOrchiectomy onlyAsymp- and disease free at 2.5 yearsUse of Birmingham Vasculitis Activity Score (BVAS) in determining need for systemic treatment for isolated PAN
Pastor-Navarro, 2007 [14]26/isolated testicular PANPainful bilateral testicular swellingLabs: all (-); U/S: multiple non-vascularized, heterogeneous, hypoechoic focal lesions; small reactive hydroceleAreas of intraparenchymal hemorrhage, fibrinoid necrosis, pleomorphic infiltrationRight orchiectomy, glucocorticoidAsymp- at 1 yearConcurrent bilateral testicular involvement
Tanuma, 2003 [15]40/isolated testicular PANLeft testicular pain, with recurrence on right 16 months after initial diagnosisLabs: (+) ANA; U/S: avascularity of bilateral testesFibrinoid necrosis of small and medium sized arteries with severe inflammatory infiltrate and near obstruction of arterial lumenBilateral orchiectomyAsymp- and disease free at 22 monthsRecurrent testicular PAN
Mukamel, 1995 [16]28, 35/isolated testicular PANRight painful testicular swelling and massLabs: all (-); U/S: intratesticular hyper- and hypoechogenic areas (1); normal with hydrocele (1)Intratesticular hemorrhage and fibrosis, segmental fibrinoid necrosis, thrombosis, and perivascular fibrosis of small arteries; some aneurysmal dilatationOrchiectomy onlyAsymp- and disease free at 2 - 3 yearsIsolated testicular PAN
Warfield, 1994 [17]19/isolated testicular PANLeft testicular pain and swelling with recurrence in right testicle 12 months laterLabs: elevated ESR and CRP, (-) ANCA; U/S: heterogeneous echo pattern including areas suggestive of cystic changeGross: scattered areas of hemorrhage of lower pole of testis; Microscopic: patchy, necrotizing vasculitis affecting medium and small arteries; fibrinoid necrosis of walls and transmural infiltrate of PMNs and lymphocytesLeft orchiectomy + cyclophosphamide × 1month, azathioprine and oral prednisone with short-term increase after recurrence on right side, long-term low dose prednisoloneRecurrence of sx at 12 months; asymp- and disease free at 18 monthsRecurrence of sx while on systemic therapy
Fleischmann, 2007 [18]21/isolated testicular PAN + metastatic mixed germ cell tumorLeft painful scrotal swellingLabs: N/A; U/S: enlarged left epididymis and calcificationsMixed germ cell tumor of left testis; circumferential, transmural fibrinoid necrosis, inflammatory infiltrates composed of neutrophils and monocytes in or around the walls of small to medium-sized arteriesOrchiectomy onlyAsymp- and disease free at 2 yearsIsolated PAN presenting with mixed germ cell tumor