Zhao Cathy Y, Chiang Yi Zhen, Murrell Dedee F
Department of Dermatology, St. George Hospital, Sydney, New South Wales, Australia.
Department of Medicine, University of New South Wales, Sydney, New South Wales, Australia.
Pediatr Dermatol. 2016 Jul;33(4):367-74. doi: 10.1111/pde.12859. Epub 2016 Apr 18.
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.
我们旨在更好地了解新生儿自身免疫性水疱病(AIBD)的发病机制、临床特征、预后及治疗。我们检索了医学文献数据库(Medline)、荷兰医学文摘数据库(Embase)、美国国立医学图书馆生物医学期刊数据库(PubMed)、拉丁美洲和加勒比健康科学文献数据库以及已识别文章的参考文献列表。纳入标准为1946年至2014年12月期间以任何语言发表的文章。排除标准为年龄大于4周且未确诊AIBD。我们共识别出51例新生儿AIBD:34例天疱疮(31例寻常型天疱疮[PV],3例落叶型天疱疮[PF])和17例类天疱疮疾病(9例大疱性类天疱疮[BP],5例线状IgA大疱性皮病[LABD],1例BP合并LABD,1例获得性大疱性表皮松解症,1例大疱性系统性红斑狼疮)。类天疱疮疾病的男性优势高于天疱疮(男:女比例为4.6:1,天疱疮为1:1.06)(p = 0.004)。天疱疮在出生时出现的比例(79.4%)高于类天疱疮疾病(29.4%)(p = 0.008)。最常受累的部位是躯干(63.0%),其次是头颈部(60.9%)。32.6%的病例累及黏膜(天疱疮为27.6%,类天疱疮疾病为41.6%)。仅33.3%的病例使用了全身治疗,75.5%的病例在3周内病情得到控制。大多数PV、PF和BP病例(但LABD病例除外)报告有母体疾病。在天疱疮病例中,75.0%的母亲患有活动性疾病,25.0%病情得到控制。任何严重程度的PV、PF和PG孕妇均可被动传递自身抗体导致新生儿AIBD。类天疱疮疾病更易在出生后出现,且可能男性更为多见。LABD的表现可能与所有其他AIBD不同。