Polyarteritis Nodosa in Childhood: Recognition of Early Dermatologic Signs May Prevent Morbidity

Beckum, Kathleen; Kim, J B A David; Kelly, David R.; Weiser, Peter; M.D., M.P.H Diego A. Lara; Theos, Amy

doi:10.1111/pde.12207

Cited by 6 publications

(7 citation statements)

References 11 publications

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“…In children, Ozen et al reported gastrointestinal symptoms in 24% of 63 children with PAN [ 6 ]. Another study evaluated the cardiac findings of 15 children with childhood PAN [ 2 ]. The most common findings were diminished left ventricular systolic function and mild mitral and/or tricuspid valve regurgitation.…”

Section: Discussionmentioning

confidence: 99%

“…The clinical spectrum is highly variable where it can be systemic or localized to one organ or system [ 1 ]. PAN occurs primarily in middle-aged adults but appears even more rarely in children where both girls and boys are affected equally [ 2 ]. It occurs in different forms as cutaneous (the most common form), classic, systemic, or microscopic.…”

Section: Introductionmentioning

confidence: 99%

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Myocardial Infarction in a 7-Year-Old Girl with Polyarteritis Nodosa

Bayazeed

Felimban

Alsaiad

et al. 2022

Case Reports in Rheumatology

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Polyarteritis nodosa (PAN) is a rare systemic vasculitis that affects small to medium-sized arteries. It could affect any organ including the heart. However coronary artery involvements are very rare. We describe a young girl who presented following a histopathological diagnosis of PAN with acute chest pain, high serum troponin, and progressive ischemic changes in the electrocardiogram (ECG). Induction of remission of her disease was done with six-moths Cyclophosphamide infusions and pulse corticosteroids. In addition to anticoagulation and dual antithrombotic therapy, the disease remission was maintained with mycophenolate mofetil which helps in the recovery of coronary disease. Our case illustrates the serious cardiac involvement of PAN in a child that responded to intensive management.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Myocardial Infarction in a 7-Year-Old Girl with Polyarteritis Nodosa

Bayazeed

Felimban

Alsaiad

et al. 2022

Case Reports in Rheumatology

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“…PAN with ruptured aneurysms is a rare but life-threatening condition in children. Several studies have revealed high mortality rates of PAN in children who presented with acute abdomen despite the administration of combined medical and surgical treatment 7,8. Approximately 50% of the patients with systemic PAN have GI involvement ranging from mild to severe manifestations 1.…”

Section: Discussionmentioning

confidence: 99%

<p>Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab</p>

Lerkvaleekul

Treepongkaruna

Ruangwattanapaisarn

et al. 2019

BTT

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Systemic polyarteritis nodosa (PAN) is a rare form of necrotizing vasculitis in children. Recurrent episodes of abdominal aneurysm ruptures are uncommon and life-threatening condition in children. Failures of response to immunosuppressive medications and radiological intervention also lead to high mortality. Some reports suggested that tumor necrosis factor (TNF) might have role in the inflammation of this disease. After an English-language literature review, this is the first case report in children of recurrent abdominal-ruptured aneurysms with a failure of conventional therapy but successfully treated with anti-TNF-α monoclonal antibody. We herein describe a 9-year-old girl who presented with chronic abdominal pain, hypertension, and massive lower gastrointestinal bleeding. The disease was refractory to conventional treatment, including administration of a corticosteroid, cyclophosphamide, and intravenous immunoglobulin, and recurrent-ruptured aneurysms developed in the gastrointestinal tract. Arterial embolization during angiography resulted in temporary improvement of the gastrointestinal bleeding. Infliximab, a chimeric anti-tumor necrosis factor-α monoclonal antibody, was initiated and resulted in disease remission with resolution of the gastrointestinal bleeding and abdominal pain. Anti-TNF therapy might be another treatment option for refractory disease to prevent ongoing inflammation that could lead to aneurysmal dilatation or even rupture. However, early recognition of refractory disease and aggressive treatment in the early course of the disease are crucial to reduce morbidity and mortality.

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“…Criteria for diagnosing childhood PAN were published recently by the European League Against Rheumatism/Paediatric Rheumatology International Trials Organisation/Paediatric Rheumatology European Society (see table 1). 5 9…”

Section: Discussionmentioning

confidence: 99%

Atypical Henoch-Schonlein purpura? Consider polyarteritis nodosa!

Braungart¹,

Campbell

Besarovic

2014

BMJ Case Reports

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We describe the case of a previously healthy 12-year-old boy admitted to a tertiary paediatric centre with the clinical diagnosis of Henoch-Schonlein purpura (HSP). Symptoms on admission included a generalised rash, colicky abdominal pain, hypertension, proteinuria and fresh rectal bleeding. Abdominal pain and distension worsened and serial ultrasounds suggested bowel ischaemia. He underwent repeat laparotomy and bowel resection, with slow improvement after the second laparotomy. The severity of systemic involvement (gastrointestinal, cardiac, renal and skin) made the initial diagnosis of HSP questionable. Immunohistochemistry of skin biopsies was negative for HSP. Histopathology of the bowel specimen revealed features of necrotising small and medium vessel vasculitis in keeping with polyarteritis nodosa.

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Polyarteritis Nodosa in Childhood: Recognition of Early Dermatologic Signs May Prevent Morbidity

Cited by 6 publications

References 11 publications

Myocardial Infarction in a 7-Year-Old Girl with Polyarteritis Nodosa

Myocardial Infarction in a 7-Year-Old Girl with Polyarteritis Nodosa

<p>Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab</p>

Atypical Henoch-Schonlein purpura? Consider polyarteritis nodosa!

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