Polyarteritis nodosa isolated to muscles-A case series with a review of the literature

Ganeshanandan, Luckshman; Brusch, Anna; Dyke, Jason; McLean‐Tooke, Andrew

doi:10.1016/j.semarthrit.2019.12.002

Cited by 10 publications

(5 citation statements)

References 15 publications

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“…The high incidence of myalgia is in line with the common involvement of intramuscular arteries and the peripheral nervous system in PAN [7] [12]. Patients with myositis-associated PAN usually present with symmetrical, proximal muscle weakness prominently in the lower extremities [7] [13] [14] [20] [21].…”

Section: Discussionmentioning

confidence: 99%

A Case of Polyarteritis Nodosa with Positive Anti-Phospholipid Antibodies Presenting with Multifocal Myositis

Thu,

Le,

Hoang

et al. 2024

Preprint

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Background: Polyarteritis nodosa (PAN) is a rarely necrotizing vasculitis in childhood and is characterized by the inflammation of small and medium vessels affecting multiple organs. Although the presence of antiphospholipid (aPL) antibodies in PAN has been documented, there is limited data on the prevalence and understanding of pathogenesis and management for such co-incidence. Case presentation: We herein reported that a 7-year-old boy without significant past medical history presented initially with high-grade fever, painful subcutaneous nodules, and ankle arthritis for 14 days prior to hospitalization. Biological findings revealed increased acute inflammatory biomarkers and no evidence of infection. The deep skin biopsy taken from the subcutaneous nodule proved leukocytoclastic medium-sized vasculitis suggestive of the PAN diagnosis. He eventually achieved a complete response with the use of intravenous corticosteroids (2 mg/kg/day) and subcutaneous methotrexate (15 mg/m2 body surface area (BSA)/week). However, the patient developed painfulness of bilateral proximal muscles and new-onset subcutaneous nodules of lower limbs after 1-week maintenance. Though the creatine phosphokinase (CPK) level was normal, the magnetic resonant imaging (MRI) showed multiple foci myositis of bilateral gluteal and femur regions. The serum level of D-dimer was remarkably elevated, and the lupus anticoagulant was positive. A combination of subcutaneous enoxaparin and pulsed cyclophosphamide (500 mg/m2 BSA) and three-day methylprednisolone (30 mg/kg/day) have contributed to a favorable outcome in this case. She further sustained remission on maintenance of gradually tapering doses of oral prednisolone and methotrexate. Conclusions: We describe a case of PAN with the presence of aPL antibody manifesting with multifocal myositis. It is recommended that general testing for aPL antibodies should be undergone in patients with PAN, as well as other systemic vasculitis. Despite unusual co-incidence, aPL antibodies might worsen systemic vasculitis through thrombotic events, which clinicians should consider adequate coagulant therapeutics besides immunosuppressors.

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Section: Discussionmentioning

confidence: 99%

A Case of Polyarteritis Nodosa with Positive Anti-Phospholipid Antibodies Presenting with Multifocal Myositis

Thu,

Le,

Hoang

et al. 2024

Preprint

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“…In addition, the initial laboratory results showed an elevation of CPK level, which is an atypical finding in PAN, making the diagnosis even more challenging. [3,8]…”

Section: Discussionmentioning

confidence: 99%

“…In addition, the initial laboratory results showed an elevation of CPK level, which is an atypical finding in PAN, making the diagnosis even more challenging. [3,8] The pathogenesis of peripheral neuropathy in PAN is focal or multifocal axonal ischemic neuropathy caused by the occlusion of the vasa nervorum. [9] This is consistent with the findings of axonal loss-type sciatic neuropathy observed in the patient's electrodiagnostic tests.…”

Section: Discussionmentioning

confidence: 99%

Polyarteritis nodosa initially presenting as concomitant peripheral neuropathy and myositis in unilateral limb: A case report

2023

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Rationale: We report the case of a patient who initially presented with peripheral neuropathy and myositis without typical organ involvement, such as the kidneys, skin, or gastrointestinal system, but was ultimately diagnosed with polyarteritis nodosa (PAN). Patient concerns: A 62-year-old man presented with radicular pain in his right lower extremity. One week later, he complained of right ankle motor weakness and pain in the right posterior thigh, which led to admission. After 6 weeks of hospitalization, he newly experienced pain in his right testicle and anterior thigh. Diagnosis: The patient was initially diagnosed with polymyositis combined with sciatic neuropathy using magnetic resonance imaging, electrodiagnostic tests, and muscle biopsy. However, with the emergence of other systemic symptoms such as testicular pain, vasculitis was suspected, and the patient was reclassified as PAN using the 2007 European Medicines Agency algorithm and the American College of Rheumatology criteria. Interventions: The patient was treated with glucocorticoids for more than 6 months, and antiviral medication was prescribed to prevent hepatitis B virus reactivation. Outcomes: The patient’s radicular pain and pain in the right anterior and posterior thighs and testicle improved, and there were no signs of recurrence. Lessons: In patients presenting with radicular and focal muscle pain, it is crucial to consider the potential for PAN, as observed in this case report.

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“…In clinical practice, the diagnosis of vasculitis is often delayed when patients present with primary complaints of extremity symptoms (muscular pain, edema, numbness, etc.) without evident organ manifestations of vasculitis, such as skin, kidney, or lung lesions 21 . For patients exhibiting clinical symptoms of their extremities, muscle MRI offers a noninvasive option, and MRI studies can be easily conducted.…”

Section: Introductionmentioning

confidence: 99%

“…without evident organ manifestations of vasculitis, such as skin, kidney, or lung lesions. 21 For patients exhibiting clinical symptoms of their extremities, muscle MRI offers a noninvasive option, and MRI studies can be easily conducted. In addition, muscle MRI of the extremities can illustrate a relatively broad area and is suitable for detecting affected vessels.…”

mentioning

confidence: 99%

Comparison of characteristics of muscle magnetic resonance imaging findings in patients with antineutrophilic cytoplasmic antibody‐associated vasculitis and polyarteritis nodosa

Ito,

Yoshida,

Fukuda

et al. 2024

Int J of Rheum Dis

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AimThis study aimed to analyze the muscle magnetic resonance imaging (MRI) findings of patients with antineutrophilic cytoplasmic antibody‐associated vasculitis (AAV) and polyarteritis nodosa (PAN) presenting with clinical symptoms in the extremities.MethodsRetrospective analysis was conducted on short tau inversion recovery MRI findings, with a focus on intramuscular vessels displaying abnormal perivascular signals, in 22 and eight patients with AAV and PAN, respectively. The number per unit area (4 cm2) and diameter of abnormal vessels on muscle MRI were compared between patients with AAV and those with PAN. Cut‐off values, clinical sensitivity, and specificity for these indices were calculated from the receiver operating characteristic curves to distinguish between AAV and PAN, and the relationship between the indices and clinical findings in AAV was analyzed.ResultsThe number of abnormal vessels per unit area was significantly higher in AAV compared to PAN (p < .05). Additionally, the diameter of the abnormal vessels was significantly higher in PAN than in AAV (p < .05). The presence of >6.44 abnormal vessels per unit area or ≤3.61 mm diameter of abnormal vessels was able to predict AAV (sensitivity, 0.955; specificity, 0.625). AAV patients with peripheral neuropathy exhibited a significantly higher number of abnormal vessels per unit area than those without peripheral neuropathy (p < .05).ConclusionsMuscle MRI can detect small‐ to medium‐vessel vasculitis and be a valuable tool for distinguishing between patients with AAV and PAN experiencing clinical symptoms in the extremities.

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Polyarteritis nodosa isolated to muscles-A case series with a review of the literature

Cited by 10 publications

References 15 publications

A Case of Polyarteritis Nodosa with Positive Anti-Phospholipid Antibodies Presenting with Multifocal Myositis

A Case of Polyarteritis Nodosa with Positive Anti-Phospholipid Antibodies Presenting with Multifocal Myositis

Polyarteritis nodosa initially presenting as concomitant peripheral neuropathy and myositis in unilateral limb: A case report

Comparison of characteristics of muscle magnetic resonance imaging findings in patients with antineutrophilic cytoplasmic antibody‐associated vasculitis and polyarteritis nodosa

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