Multiple Colonic Ulcers Caused by Churg–Strauss Syndrome in a 15-Year-Old Girl

Lin, Tzu‐Lung; Wang, Chrong‐Reen; Liu, M.-F.; Chen, P.-C.; Shan, Yuheng; Jin, Ying; Chien, Chia-Hung

doi:10.1007/pl00011209

Cited by 19 publications

(14 citation statements)

References 10 publications

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“…Moreover, such severe organ involvements are attributable to a worse prognosis and mortality (Zwerina et al 2008). Like our patient, some cases exhibiting multiple colonic ulcers associated with CSS have been reported (Shimamoto et al 1990;Berarducci et al 1996;Lin et al 2001). However, the treatment of pediatric patients with CSS, especially in severe cases, remains challenging (Ikemoto et al 2001;Boyer et al 2006;Zwerina et al 2008;Domircin et al 2010).…”

Section: Discussionsupporting

confidence: 48%

Successful Multidrug Treatment of a Pediatric Patient with Severe Churg-Strauss Syndrome Refractory to Prednisolone

Watanabe

Aizawa-Yashiro

Tsuruga

et al. 2011

Tohoku J. Exp. Med.

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Section: Discussionsupporting

confidence: 48%

Successful Multidrug Treatment of a Pediatric Patient with Severe Churg-Strauss Syndrome Refractory to Prednisolone

Watanabe

Aizawa-Yashiro

Tsuruga

et al. 2011

Tohoku J. Exp. Med.

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“…Although more commonly seen among adults, CSS can present in childhood. [15][16][17] This syndrome is characterized by asthma, peripheral eosinophilia, and vasculitis and is associated with autoantibodies to perinuclear antineutrophil cytoplasmic antibody, as well as cutaneous and systemic granulomas. Palpable purpura, tender subcutaneous nodules, and cutaneous infarctions are more often the associated skin findings in CSS, whereas patients present with bullae and vesicular lesions in Wells' syndrome.…”

Section: Discussionmentioning

confidence: 99%

Bullous “Cellulitis” With Eosinophilia: Case Report and Review of Wells' Syndrome in Childhood

Gilliam¹,

Bruckner

Howard³

et al. 2005

Pediatrics

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ABSTRACT. A 1-year-old girl presented with acute onset of edematous erythematous plaques associated with bullae on her extremities and accompanied by peripheral eosinophilia. She was afebrile, and the skin lesions were pruritic but not tender. The patient was treated with intravenously administered antibiotics for presumed cellulitis, without improvement. However, the lesions responded rapidly to systemic steroid therapy. On the basis of lesional morphologic features, peripheral eosinophilia, and cutaneous histopathologic features, a diagnosis of Wells' syndrome was made. Wells' syndrome is extremely rare in childhood, with 27 pediatric cases reported in the literature. Because it is seen so infrequently, there are no specific guidelines for evaluation and management of Wells' syndrome among children. W ells' syndrome, or eosinophilic cellulitis, is a rare, recurrent, inflammatory dermatosis of unknown pathogenesis. In 1971, Wells 1 described 4 patients with an acute pruritic dermatitis clinically resembling bacterial cellulitis but with histopathologic findings characterized by dermal eosinophilia, phagocytic histiocytes, and the presence of flame figures. He initially called this syndrome recurrent granulomatous dermatitis with eosinophilia but later simplified the name to eosinophilic cellulitis. 2 Wells' syndrome is seen more commonly among adults but has been observed among children. Some hypothesize that this syndrome may represent a hypersensitivity response to a circulating antigen. 2 Associated precipitants include insect bites, medication reactions, recent immunization, myeloproliferative disorders, malignancies, and infections. We describe a case of a young child with no identifiable triggering factors, and we review the evidence for evaluation and management of these pediatric cases. CASE REPORTA previously healthy, 1-year-old girl presented with acute onset of edematous erythematous plaques, with associated bullae, on her lower extremities and left arm (Fig 1). These lesions were pruritic but not painful, and the patient was afebrile. Her parents denied a history of insect bites, ingestion of medications, trauma, or other intercurrent illness. The patient's most recent immunizations had been received 3 months earlier. The patient did not have a history of asthma, and there was no family history of asthma or atopic disease.The patient was admitted with presumed bacterial cellulitis and was treated with intravenously administered oxacillin, without improvement. Her laboratory studies were significant for an elevated white blood cell count of 30 ϫ 10 9 cells per L, with peripheral eosinophilia of 48%. After the patient failed to respond to systemically administered antibiotics, examination of vesicle fluid was performed and revealed numerous eosinophils. Subsequently, the diagnosis of probable Wells' syndrome was made. Oral steroid therapy was started at 2 mg/kg, and the patient's cutaneous symptoms improved within 24 hours, leaving residual erythema and hyperpigmentation (Fig 2). Five days after the i...

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“…Sometimes the involvement of the GIT occurs in isolation as a limited intestinal form of CSS. The clinical manifestations are generally related to vasculitis, eosinophilic gastroenteritis, appendicitis, or rectocolitis (Lin et al 2001). Abdominal pain, nausea, vomiting, and diarrhea are the main signs and symptoms which may develop into an acute surgical abdomen because of intestinal ischemia, causing infarction or perforation.…”

Section: Digestive Manifestationsmentioning

confidence: 99%