Infantile bullous pemphigoid treated with intravenous immunoglobulin therapy

Sugawara, Nobuyuki; Nagai, Yayoi; Matsushima, Yoichiro; Aoyama, Kumi; Ishikawa, Osamu

doi:10.1016/j.jaad.2007.08.005

Cited by 32 publications

(25 citation statements)

References 28 publications

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“…Although IBP usually responds well to first‐line therapy with systemic corticosteroids, resistant cases have been reported. A literature search revealed anecdotal cases of IBP treated using IVIG (Table ) . Variable dosages (200 mg/kg/day to 1 g/kg/day), infusion courses (2‐ to 5‐day courses), and intervals (2–5 wks) were used, probably reflecting the heterogeneity of institutional or individual preferences regarding IVIG treatment in general.…”

Section: Discussionmentioning

confidence: 99%

“…The IVIG dosing scheme of the current case (400 mg/kg/day for 5 consecutive days per infusion cycle) corresponds to the high‐dose regimen employed for the treatment of bullous pemphigoid and other autoimmune bullous diseases in adults. Similar regimens were also preferred in other cases of IBP . In the majority of the reviewed cases, clinical remission was obtained after the introduction of IVIG, although in two cases rituximab had to be added.…”

Section: Discussionmentioning

confidence: 99%

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Infantile Bullous Pemphigoid Treated Using Intravenous Immunoglobulin: Case Report and Review of the Literature

Tekin

Yücelten

2015

Pediatric Dermatology

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We report a 5-month-old girl diagnosed with bullous pemphigoid who initially did not respond to systemic corticosteroids and dapsone but rapidly improved after the addition of intravenous immunoglobulin (IVIG) infusions. A literature search revealed anecdotal cases of infantile bullous pemphigoid treated with IVIG, although variable treatment regimens were used, and some resistant cases required additional medications such as rituximab for clinical remission.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Infantile Bullous Pemphigoid Treated Using Intravenous Immunoglobulin: Case Report and Review of the Literature

Tekin

Yücelten

2015

Pediatric Dermatology

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“…Other treatments that have been used include potent topical steroids alone, particularly in childhood localized vulvar disease . In refractory cases, IVIg and rituximab have been used successfully …”

Section: Discussionmentioning

confidence: 99%

Childhood bullous pemphigoid: a report of three cases in China

Yang

Yan

et al. 2015

Int J Dermatology

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“…Literature review revealed only three additional cases of IBP treated with IVIG. 2,5,6 In conclusion, we describe a case of IBP successfully treated with IVIG therapy, which could be considered as a possible treatment for patients resistant to conventional corticosteroid therapy.…”

mentioning

confidence: 90%

“…IBP is quite rare. 2 Furthermore, Waisbourd-Zinman et al compared BP in infancy with childhood BP, and reported significantly higher predisposition for acral involvement in IBP. However, no major differences regarding laboratory results, treatments and prognosis were found.…”

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confidence: 99%

Infantile bullous pemphigoid successfully treated with intravenous immunoglobulin therapy

et al. 2017

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Bullous pemphigoid (BP) is the most common acquired autoimmune bullous dermatosis, and is extremely rare in children and infants. 1 We report a case of a child with infantile BP (IBP) that was successfully treated with intravenous immunoglobulin (IVIG) therapy.A 4-month-old boy presented with erythematous skin lesions and multiple vesicles on his hands and feet. He had originally been seen by his paediatrician, but as the erythematous lesions and blisters continued to develop, he was referred to us. He was previously healthy, and Nikolsky sign was absent. His medical history was unremarkable. DPT-IPV (combined diphtheria, Bordetella pertussis, tetanus and poliovirus), Haemophilus influenza B and Pneumoccocus immunizations had been given 10 days before his visit to the paediatrician. There was no parental history of skin blistering.Physical examination revealed urticarial plaques with tense bullae and small vesicles on the patient's arms, palms and soles. There were urticarial plaques with erosions on his face, trunk ( Fig. 1a) and limbs (Fig. 1b,c). Mucous membranes were not involved, and the rest of the physical examination was unremarkable.Histopathological examination showed a subepidermal bulla containing eosinophils (Fig. 2a,b). Direct immunofluorescence (DIF) demonstrated linear deposition of IgG (Fig. 2c) and C3 (Fig. 2d) along the dermoepidermal junction. ELISA of the patient's serum demonstrated elevated IgG autoantibodies against the NC16a domain of BP antigen 180 (BP180; also termed BP antigen 2 or type XVII collagen) at 126 (normal range: < 9).The child was diagnosed as having IBP and was initially treated with topical and oral corticosteroids (prednisolone 1 mg/kg/day), but the erythema and blisters continued to spread, so the oral prednisolone dose was increased to 2 mg/kg daily and erythromycin 40 mg/kg daily was started. However, this produced no improvement, and adverse effects (AEs) began to develop, such as moon face and impaired liver function. Therefore, a 1-day course of intravenous immunoglobulin (IVIG) therapy 2 g/kg/day was initiated. Five days after the IVIG therapy, no new lesions had developed. Recurrence of blistering occurred 2 weeks after first IVIG therapy and a second course of IVIG (2 g/kg/day) was given, which resulted in significant improvement. A third and fourth course of IVIG (1 g/kg/day) were given, and the dose of prednisolone was gradually tapered to 0.1 mg/kg/day (Fig. 2e). None of the potentially serious AEs of IVIg, such as anaphylaxis or renal failure, were experienced. During the subsequent 2-year follow-up period, the patient has not had a recurrence. IBP is quite rare. 2 Furthermore, Waisbourd-Zinman et al. compared BP in infancy with childhood BP, and reported significantly higher predisposition for acral involvement in IBP. However, no major differences regarding laboratory results, treatments and prognosis were found.

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Infantile bullous pemphigoid treated with intravenous immunoglobulin therapy

Cited by 32 publications

References 28 publications

Infantile Bullous Pemphigoid Treated Using Intravenous Immunoglobulin: Case Report and Review of the Literature

Infantile Bullous Pemphigoid Treated Using Intravenous Immunoglobulin: Case Report and Review of the Literature

Childhood bullous pemphigoid: a report of three cases in China

Infantile bullous pemphigoid successfully treated with intravenous immunoglobulin therapy

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