Polyarteritis Nodosa Presenting with Bilateral Testicular Swelling and Complicated by Unilateral Facial Nerve Palsy

Yuminaga, Yuigi; Richards, Bethan; Rasiah, K.; Thanigasalam, Ruban; Atmore, Bryn; Laurent, R

doi:10.4111/kju.2011.52.5.364

Cited by 6 publications

(8 citation statements)

References 8 publications

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“…Cranial nerve involvement in PAN is a rare manifestation, being less frequent than the central nervous sys-tem involvement and peripheral neuropathy. According to our literature review, the most frequent cranial nerves involved in PAN are the optic (II), oculomotor (III) and abducent (IV) [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21]. We did not find any case report with involvement of olfactory, glossopharyngeal, vagus, accessory or hypoglossal nerves in patients with PAN.…”

Section: Discussionmentioning

confidence: 90%

See 1 more Smart Citation

Oculomotor nerve palsy, an unusual onset of polyarteritis nodosa

Martins¹,

Pinheiro²,

Vedor³

et al. 2023

Reumatologia

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IntroductionCranial nerve involvement in polyarteritis nodosa(PAN) is underrecognized and rarely reported. The aim of this article is to review the available literature and present an example of oculomotor nerve palsy in the course of PAN.Material and MethodsEvaluation of texts describing the analyzed problem using the terms “polyarteritis nodosa”, “nerve”, “oculomotor”, “cranial nerve” and “cranial neuropathy” for searching the PubMed database was done. Only full-text articles in English language with titles and abstracts were included in the analysis. As a guideline for the analysis of articles, the methodology described in the Principles of Individual Patient Data systematic reviews (PRISMA-IPD) was used.ResultsAfter screening articles only 16 reported cases of PAN with cranial neuropathy were included in the analysis. In 10 the cranial neuropathy was reported as the initial manifestation of PAN with optic nerve involvement as the most frequent (62.5%); among these cases the oculomotor nerve was in-volved in 3 cases. Treatment with glucocorticosteroids and cyclophosphamide was the most com-mon.ConclusionsAlthough cranial neuropathy, especially oculomotor nerve palsy is a rare first neurological manifesta-tion of PAN, this clinical problem should be considered in the differential diagnosis. Especially patients with peripheral neuropathy, general symptoms, skin lesions and hepatitis B virus infection should be evaluated for cranial nerve involvement in the course of vasculitis. In the case of unclear involvement of the cranial nerves, PAN should also be considered in the differential diagnosis as the cause of symptoms and the first manifestation of the disease.

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Section: Discussionmentioning

confidence: 90%

“…1). Each case is described in Table I [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21]. In 10 of the 16 patients, cranial neu-ropathy was the initial manifestation, as in our case.…”

Section: Systematic Literature Reviewmentioning

confidence: 83%

Oculomotor nerve palsy, an unusual onset of polyarteritis nodosa

Martins¹,

Pinheiro²,

Vedor³

et al. 2023

Reumatologia

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“…Testicular pain as a presenting feature of PAN is rare, and testicular symptoms are most often associated with signs of multisystem involvement (53)(54)(55)(56)(57)(58)(59)(60)(61). Many instances of testicular involvement in PAN are asymptomatic (62).…”

Section: Discussionmentioning

confidence: 99%

“…In these individuals, the diagnosis of PAN is often made by radical orchiectomy, although testicular biopsy is sufficient for a tissue diagnosis. Sometimes, this disease remains isolated to the testes, although this is very rare and investigations for signs of systemic involvement should be performed (61). Orchiectomy is usually sufficient for the treatment of isolated testicular PAN, which carries a more favorable prognosis than systemic disease.…”

Section: Discussionmentioning

confidence: 99%

Evolving Chest Pain in a Young Male Patient

White

Cunha

2022

Arthritis Care & Research

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“…4 When hemorrhage and necrosis are seen in a testis that also has damaged blood vessels (testicular vasculopathy) there may be concern for systemic vasculitis, even though most reports of "testicular vasculitis" are said to be of "limited" type without clinical evidence of systemic disease, [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20] and it is distinctly rare for true systemic vasculitis to initially present with findings confined to the testis. 5,6,[21][22][23][24][25][26][27][28][29][30][31] We herein report our experience with 30 cases of testicular hemorrhage and necrosis with associated vasculopathy to bring attention to this pseudoneoplastic condition and also to investigate its possible etiology and relationship with systemic vasculitis. We provide evidence supporting that many of these cases are not due to true vasculitis but are secondary to chronic intermittent torsion.…”

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confidence: 99%

Testicular Hemorrhage, Necrosis, and Vasculopathy

Kao

Zhang

Ulbright

2014

American Journal of Surgical Pathology

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We report 30 cases of testicular hemorrhage and/or necrosis with associated vascular damage that caused clinical concern for a neoplasm and that raised the question of a vasculitis syndrome on pathologic examination. The patients were 12 to 66 years old (median, 33 y) and presented with pain (n=15), mass (n=12), or both (n=2); 1 case had no available clinical information. Ultrasonographic interpretations included a neoplasm in the differential diagnoses in 14 of 18 cases in which this information was available, and most (n=24) had orchiectomy because of this possibility. Only 4 were clinically suspected to represent testicular infarction. Circumscribed, hemorrhagic lesions occurred in 10 cases, less demarcated hemorrhagic foci in 5, and discrete nodules or ill-defined foci of varying color and consistency in the remainder. No clear testicular lesion was described in 2, with 1 of these having a "dusky" appearance. On microscopic examination all but 1 case showed damaged blood vessels (vasculopathy), with either associated hemorrhage/hematoma (n=24) and/or areas of parenchymal necrosis (n=21). One case showed only segmental tubular atrophy with interstitial inflammation and vasculopathy; no infarct or hemorrhage was identified. A variety of vascular changes was identified, including prominent intimal thickening in arteries (n=22) and fibrinoid change in both arteries (n=5) and vessels of indeterminate type (n=8). Medial fibrosis was present in veins (n=12) and vessels of indeterminate type (n=4), whereas thrombi (remote, recanalized, and/or recent) occurred in arteries (n =7), veins (n=9), and vessels of indeterminate type (n=11). Dilated, blood-filled vessels were present in the testis and/or paratestis in 15 cases. In addition, 7 cases showed arteriolar hyalinization, and 19 had inflammation of blood vessels. The latter was lymphohistiocytic and mostly light but occasionally prominent (n=5). Interstitial inflammation was seen adjacent to damaged testicular parenchyma in all 30 cases. Clinical follow-up in 20 patients (4 to 131 mo, mean 38 mo) showed no evidence of recurrence in the contralateral testis or later development of systemic vasculitis. The histologic findings were compared with those in 11 orchiectomies resected for clinical acute torsion. All clinical acute torsion cases showed both parenchymal and fibrinoid vascular necrosis, and 10 had hemorrhage/hematoma; they lacked vasculitis, interstitial inflammation, and chronic vascular changes. Testicular vasculopathy, characterized by acute and chronic vascular injury, commonly occurs in testes with parenchymal hemorrhage and necrosis that clinically mimic a tumor. It shares the acute features of recent torsion but also has findings indicative of chronic injury. Testicular vasculopathy is most likely a result of chronic intermittent torsion that leads to localized hemorrhage/necrosis and should be distinguished from cases of systemic vasculitis given the significantly different clinical implications.

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Polyarteritis Nodosa Presenting with Bilateral Testicular Swelling and Complicated by Unilateral Facial Nerve Palsy

Cited by 6 publications

References 8 publications

Oculomotor nerve palsy, an unusual onset of polyarteritis nodosa

Oculomotor nerve palsy, an unusual onset of polyarteritis nodosa

Evolving Chest Pain in a Young Male Patient

Testicular Hemorrhage, Necrosis, and Vasculopathy

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