Infantile bullous pemphigoid successfully treated with combined pulse corticosteroids and high-dose IVIG

Wawrzycki, Bartłomiej; Krasowska, Dorota; Pietrzak, Aldona; Szumiło, Justyna; Błażowski, Łukasz; Pietraszek-Mamcarz, Jolanta; Lotti, Torello

doi:10.1111/dth.12635

Cited by 3 publications

(2 citation statements)

References 9 publications

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“…Średni wiek pacjentów w momencie rozpoznania w Europie wynosi około 80 lat [5]. Niemowlęce i dziecięce odmiany pemfigoidu pęcherzowego (określane jako pemfigoid niemowlęcy i pemfigoid dziecięcy) należą do chorób niezwykle rzadkich [6]. Pierwszy przypadek pemfigoidu pęcherzowego u dziecka opisano w 1970 roku na podstawie bezpośredniej diagnostyki immunofluorescencyjnej [7], a pierwszy przypadek u niemowlęcia -w 1977 roku [7].…”

Section: Wprowadzenieunclassified

“…The mean age of patients at diagnosis in Europe is around 80 years [5]. In contrast, infantile and childhood varieties of bullous pemphigoid (described as infantile pemphigoid and childhood pemphigoid) are exceedingly rare diseases [6]. The first case of bullous pemphigoid in a child was described in 1970 based on direct immunofluorescence diagnosis [7] and the first case of bullous pemphigoid in an infant was described in 1977 [7].…”

Section: Lokalizacja Zmian Skórnychmentioning

confidence: 99%

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Pemphigoid diseases in infancy and childhood. A review of the literature

Jałowska,

Dmochowski

2023

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Bullous pemphigoid is an autoimmune blistering disease that very rarely affects children. Clinical differences in locations of skin lesions led to the distinction of infantile versus childhood pemphigoid. Mucous membranes can be affected. The first-line therapy usually consists of topical or/and systemic glucocorticosteroids. Intravenous immunoglobulin infusions can be a valuable treatment option. Prognosis of infantile pemphigoid and childhood pemphigoid is good and the duration is often limited to one year with adequate treatment. The differential diagnosis of blisters should include: linear immunoglobulin A bullous disease of childhood, epidermolysis bullosa acquisita, bullous systemic lupus erythematosus, bullous scabies and childhood infectious diseases with blisters.

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Section: Wprowadzenieunclassified

Section: Lokalizacja Zmian Skórnychmentioning

confidence: 99%

Pemphigoid diseases in infancy and childhood. A review of the literature

Jałowska,

Dmochowski

2023

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Hib-DTaP-poliovirus-vaccine

2018

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Neonatal Autoimmune Subepidermal IgG/IgA Blistering Disease With Severe Laryngeal and Esophageal Involvement: A Report of a Case and Review of the Literature

Raiber

Sezin

Sadik

et al. 2020

The American Journal of Dermatopathology

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Neonatal autoimmune subepidermal blistering disease is rare. Mucosal involvement is more common in neonatal linear immunoglobulin A (IgA) bullous dermatosis. We describe a neonate with subepidermal cutaneous blistering disease with severe laryngeal and esophageal involvement leading to acute respiratory distress. Histopathology demonstrated a subepidermal blister with neutrophils and eosinophils at the dermal base. Collagen IV was detected at the dermal floor, and direct immunofluorescence showed linear IgG, IgA, and C3 deposits at the basement membrane zone. The patient demonstrated markedly increased serum levels of anti-BP180 NC16A and anti-BP230 IgG antibodies (Abs) but failed to show anti-LAD-1 IgA Abs. His healthy mother showed serum anti-LAD-1 IgA Abs but did not show anti-BP180 and anti-BP230 Abs. The neonate responded promptly to systemic corticosteroid therapy. A review of the literature detected 11 cases of neonatal subepidermal blistering disease with linear IgA deposits. Nine of these cases demonstrated coexisting linear IgG deposits, often with C3. Respiratory compromise was present in most of the cases. Neutrophils and eosinophils were commonly present in the inflammatory cell infiltrates. Besides our case, 2 cases of neonatal IgG/IgA subepidermal blistering disease with esophageal involvement were previously described. IgA Abs were present in the sera of both cases. Anti-LAD-1 IgA Abs were detected in the mother's serum of our case alone, but IgA Abs do not cross the placenta. Our case was consistent with neonatal IgG/IgA pemphigoid. Neonatal IgG/IgA subepidermal blistering disease may be associated with severe laryngeal and esophageal involvement leading to respiratory compromise. Expedited diagnosis and prompt treatment are warranted.

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Infantile bullous pemphigoid successfully treated with combined pulse corticosteroids and high-dose IVIG

Cited by 3 publications

References 9 publications

Pemphigoid diseases in infancy and childhood. A review of the literature

Pemphigoid diseases in infancy and childhood. A review of the literature

Hib-DTaP-poliovirus-vaccine

Neonatal Autoimmune Subepidermal IgG/IgA Blistering Disease With Severe Laryngeal and Esophageal Involvement: A Report of a Case and Review of the Literature

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