Neonatal Pemphigus Vulgaris in an Infant Born to a Mother with Pemphigus Vulgaris in Remission

Fenniche, Samy; Benmously, Rym; Marrak, H.; Dhaoui, Asma; Ammar, Feiza Ben; Mokhtar, I.

doi:10.1111/j.1525-1470.2006.00195.x

Cited by 24 publications

(21 citation statements)

References 24 publications

(12 reference statements)

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“…[18] The maternal disease was not always severe; it may be mild as in our case or even inactive. [5,31,32] Although there is no prospective or cohort study defining the best drug regimen during pregnancy in PV, prednisolone and azathioprine are considered safe therapeutic options in this context. [20] Topical steroids and intralesional steroid injection (triamcinolone acetonide 10-20 mg/mL around the lesion) can be used in mild cases.…”

Section: Discussionmentioning

confidence: 98%

“…As pemphigus is a rare disease affecting mostly women of the 5 th and 6 th decades in Western countries, [2,3] studies on the association of pemphigus and pregnancy in the literature are limited to case reports and small case series not exceeding more than 47 pemphigus vulgaris (PV) cases. [1,[4][5][6][7] Accordingly, many aspects of this topic are still unclear.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Pemphigus and pregnancy: A 23-year experience

Daneshpazhooh¹,

Chams‐Davatchi²,

Valikhani³

et al. 2011

Indian J Dermatol Venereol Leprol

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Section: Discussionmentioning

confidence: 98%

Section: Introductionmentioning

confidence: 99%

Pemphigus and pregnancy: A 23-year experience

Daneshpazhooh¹,

Chams‐Davatchi²,

Valikhani³

et al. 2011

Indian J Dermatol Venereol Leprol

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“…Maternal pemphigus activity generally did not correlate with neonatal pemphigus activity. We identified multiple cases of neonatal pemphigus in children born to mothers with no or controlled pemphigus , and there have been cases of disease‐free neonates born to mothers with severe pemphigus . Also, maternal activity did not correlate with the neonate's time to disease control (Spearman ρ = 0.091).…”

Section: Discussionmentioning

confidence: 99%

Neonatal Autoimmune Blistering Disease: A Systematic Review

Zhao

Chiang

Murrell

2016

Pediatric Dermatology

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We aimed to better understand the pathogenesis, clinical features, prognosis, and treatment of neonatal autoimmune blistering diseases (AIBDs). We searched Medline, Embase, PubMed, Latin American and Caribbean Health Sciences Literature, and reference lists of identified articles. Inclusion criteria were articles published from 1946 to December 2014 in any language. Exclusion criteria were age greater than 4 weeks and no confirmed AIBD diagnosis. We identified 51 cases of neonatal AIBDs: 34 cases of pemphigus (31 pemphigus vulgaris [PV], 3 pemphigus foliaceus [PF]) and 17 cases of pemphigoid diseases (9 bullous pemphigoid [BP], 5 linear immunoglobulin A bullous dermatosis [LABD], 1 BP and LABD, 1 epidermolysis bullosa acquisita, 1 bullous systemic lupus erythematosus). Pemphigoid diseases had a higher male predominance (male:female ratio 4.6:1) than pemphigus (male:female ratio 1:1.06) (p = 0.004). Pemphigus had a higher proportion presenting at birth (79.4%) than pemphigoid diseases (29.4%) (p = 0.008). The most common sites involved were the trunk (63.0%), followed by the head and neck (60.9%). The mucosal membranes were involved in 32.6% of cases (27.6% in pemphigus, 41.6% in pemphigoid diseases). Only 33.3% used systemic therapy, and 75.5% achieved control within 3 weeks. Most PV, PF, and BP cases, but not LABDs, reported maternal disease. In pemphigus cases, 75.0% of mothers had active disease and 25.0% were in control. Pregnant women with PV, PF, and PG of any severity can passively transfer autoantibodies leading to neonatal AIBD. Pemphigoid diseases are more likely to present after birth and may be more male predominant. The presentation of LABDs may be different from that of all other AIBDs.

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“…Four instances of stillborn children and some premature deliveries have been reported for mothers with PV (2). The cause of intrauterine death has not been established.…”

Section: Discussionmentioning

confidence: 99%

A Newborn with Erythematous, Desquamative Plaques

Bodet¹,

Bassas²,

Bartralot³

et al. 2008

Pediatric Dermatology

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A healthy 10-year-old boy was evaluated for a rapidly growing dark black lesion of 4 weeks duration. The growth was located 5 mm inferior to the left labial commissure (Fig. 1). The mother admitted probing the growth with a needle on the assumption that a foreign body was present. Review of systems was noncontributory. The family history was negative for atypical nevi, melanoma, actinic keratoses, and nonmelanoma skin cancer.Physical examination showed a healthy Caucasian boy. An oval-shaped, dark black, slightly depressed 5 · 6 mm papulonodule was noted; it was indurated to intra-extraoral palpation. Dimple sign was positive. He had no in-transit lesions or submandibular or cervical adenopathy. A 6 mm punch biopsy specimen was obtained (Figs. 2-4).A 1-day-old white infant girl was admitted to the neonatal inpatient unit of our hospital with skin lesions suspected clinically to be a herpes simplex infection. She had been delivered without complications (Initial Apgar score 8 ⁄ 10; birth weight 2340 g) after an uneventful pregnancy of 41 weeks. Her mother during the second month of pregnancy had oral lesions which had been diagnosed as aphthosis. The girl was the first child of nonconsanguineous parents. Physical examination revealed erythematous and desquamative plaques involving the seborrheic areas, in particular the scalp, eyelids, neck, axillary folds and upper middle part of the back (Fig. 1).After 4 days of intravenous aciclovir, ampicillin, and gentamicin, the skin lesions had become more vegetative, more erythematous and oozing and less desquamative. Some flaccid vesicles and pustules and some skin erosions were seen on the left axillary fold and on the right shoulder (Fig. 2). Mucous membranes and the remaining skin were not involved.Cytologic studies from a vesicle, performed at admission, were reported as showing nuclear changes that suggested viral infection but not being specific for herpetic infection. Further microbiologic cultures (bacteria, virus, and fungi) were negative.Examination of a skin biopsy specimen, taken from the back, demonstrated an epidermal pattern of eosinophilic spongiosis. Within the papillary dermis, edema and an inflammatory infiltrate composed of eosinophils and neutrophilis were appreciated (Fig. 3).A 10-month-old girl was brought to our hospital because of enlarged right labia minor, which had not changed since her mother first noticed the swelling 1 week earlier. Physical examination demonstrated that the right labia minor was enlarged to 14 · 7 mm (Fig. 1). The overlying skin was slightly yellow and shiny. On general physical examination, no palpable lymphadenopathy or hepatosplenomegaly was found. Results of laboratory studies including a complete blood count, urinalysis, serum cholesterol, and triglyceride levels were normal. She was followed for 2 months without medication, but the enlarged labia minor did not change significantly in this time. Reductive resection under general anesthesia was undertaken and the tissue sent for histologic analysis (Figs. 2, 3).

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Neonatal Pemphigus Vulgaris in an Infant Born to a Mother with Pemphigus Vulgaris in Remission

Cited by 24 publications

References 24 publications

Pemphigus and pregnancy: A 23-year experience

Pemphigus and pregnancy: A 23-year experience

Neonatal Autoimmune Blistering Disease: A Systematic Review

A Newborn with Erythematous, Desquamative Plaques

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