Neonatal pemphigus vulgaris associated with mild oral pemphigus vulgaris in the mother during pregnancy

Chowdhury, M. M. U.; Natarajan,

doi:10.1046/j.1365-2133.1998.02418.x

Cited by 64 publications

(40 citation statements)

References 26 publications

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“…1,3,4 However, there is no clear correlation between the severity and pattern of maternal involvement, level of antibody titers or prematurity, and the neonatal clinical presentation. 5 Of note, in some cases of neonatal pemphigus, 3,4 including our case, DIF is positive for IgG and C3.…”

Section: Discussionmentioning

confidence: 91%

“…In previously reported cases, the infants' skin lesions completely healed within a few weeks of delivery. [4][5][6] However, if a pregnant woman is known to have pemphigus vulgaris, therapy during pregnancy should be aimed at reducing the antibody titer to lower the risk of transmission to the neonate. The most commonly used therapy is oral prednisone, but a recent report 7 showed that intravenous immunoglobulin administered at 4-week intervals throughout the pregnancy may be safe and effective.…”

Section: Discussionmentioning

confidence: 98%

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Blisters and Erosions in a Neonate

2014

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A newborn male infant was transferred to the neonatal intensive care unit for widespread vesicles and erosions evident at birth, following a spontaneous vaginal delivery complicated by fetal meconium exposure. The mother, initially unavailable for examination because she remained at the facility where she had given birth, reported via telephone that she was healthy except for a urinary tract infection for which she had taken a 7-day course of nitrofurantoin monohydrate approximately 3 to 4 weeks before delivery. She denied a history of herpesvirus infection, blistering disorder, or autoimmune disease. On physical examination, the neonate exhibited extensive erosions rimmed by purulent vesicles involving the groin, neck, scalp, fingers, and left side of the upper chest ( Figure, A and B). Mucous membranes were spared. Biopsy specimens were obtained, with results as shown in the Figure,

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Section: Discussionmentioning

confidence: 91%

Section: Discussionmentioning

confidence: 98%

Blisters and Erosions in a Neonate

2014

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“…Affected infants' skin disease resolves within weeks and may require treatment with topical antibiotics, topical corticosteroids, or supportive care only with bland emollients. 95,102 Ruach et al 96 suggest that conception be timed during a period of clinical remission with low immunofluorescence titers. The authors also recommend fetal screening with surveillance sonography, fetal movement counting, and repeat nonstress tests during the latter period of pregnancy.…”

Section: What Are the Differences Between Pemphigus Vulgaris In Childmentioning

confidence: 98%

“…100, 101 Antibody titers in the mother have ranged from 1:20 to 1:640 and several authors suggest that neither the antibody titers nor clinical severity of the mother's findings predict the severity of disease in the child. 99,102,103 All of the cases of intrauterine fetal death and growth retardation have been described in mothers with severe clinical disease. 96 This finding suggests that pathogenesis is multifactorial and that too few cases have been described to predict fetal outcome.…”

Section: What Are the Differences Between Pemphigus Vulgaris In Childmentioning

confidence: 98%

Bullous diseases: Kids are not just little people

Marathe

Morel

2015

Clinics in Dermatology

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“…Two patients have been described in the literature, including 1 patient who developed EBA on day 2 post partum, with resolution of blistering at menopause, 25 and 1 patient who had a relapse of her EBA during the first month of gestation, with marked improvement of her skin following termination of the pregnancy. 26 As far as we know, congenital EBA has not been reported previously, likely because EBA itself is an extremely rare disorder. Experience with neonatal pemphigus has shown that such vertically transmitted autoimmune blistering disease appears to be selflimited and resolves with supportive therapy.…”

Section: Report Of a Casementioning

confidence: 99%

Congenital Epidermolysis Bullosa Acquisita

Abrams

Smidt²,

Benjamin

et al. 2011

Arch Dermatol

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Background: Epidermolysis bullosa acquisita (EBA) is a rare, chronic, autoimmune bullous dermatosis that is caused by autoantibodies against the noncollagenous terminus of the ␣ chain of type VII collagen, resulting in decreased anchoring fibrils in the lamina densa. It classically presents with skin fragility and trauma-induced blisters that are particularly extensive over the distal aspect of the extremities and that heal with milia, dyspigmentation, and scarring, similar in presentation to dystrophic epidermolysis bullosa. Disease onset is typically in adulthood, although rare cases of childhood disease occur. To our knowledge, a case involving a neonate with congenital EBA has not yet been reported in the literature. We describe a newborn with transient EBA due to the passive transfer of maternal autoantibodies.Observations: A 2-day-old girl was evaluated for tense blisters and areas of denuded skin that had been present since birth. Her mother carried the diagnosis of EBA. The results of histopathologic analysis, immunofluores-cence studies, and enzyme-linked immunosorbent assay confirmed the diagnosis of neonatal EBA. The patient improved with supportive therapy and has not required systemic intervention.Conclusions: Autoimmune neonatal bullous skin disease caused by placental transfer of maternal IgG autoantibodies is rare. It has been reported in neonates born to mothers with pemphigus vulgaris, pemphigus foliaceus, and gestational pemphigoid. To our knowledge, congenital EBA has not been previously reported. Vertically acquired congenital autoimmune blistering disorders appear to be self-limited and resolve with supportive therapy, concomitant with the presumed clearance of maternal autoantibodies from the neonate's circulation.

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Neonatal pemphigus vulgaris associated with mild oral pemphigus vulgaris in the mother during pregnancy

Cited by 64 publications

References 26 publications

Blisters and Erosions in a Neonate

Blisters and Erosions in a Neonate

Bullous diseases: Kids are not just little people

Congenital Epidermolysis Bullosa Acquisita

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