ABSTRACT
Dichorionic diamniotic twins were born at 37 weeks of gestation by cesarean section to a 34-year-old primigravid Japanese woman because the first twin was in breech presentation. The mother had been diagnosed with pemphigus vulgaris prior to her pregnancy. In addition to a high antidesmoglein 3 autoantibody titer, flaccid bullae and erosions on both of the twins' lips and in their oral cavities at 13 days of age led to the diagnosis of neonatal pemphigus vulgaris. This case highlights the need for awareness that pemphigus vulgaris may not occur immediately after birth.
Subject(s)
Infant, Newborn, Diseases/immunology , Pemphigus/congenital , Pemphigus/immunology , Pregnancy Complications/immunology , Prenatal Exposure Delayed Effects/immunology , Twins , Adult , Autoantibodies/immunology , Desmoglein 3/immunology , Female , Humans , Infant, Newborn , Male , Maternal-Fetal Exchange , PregnancyABSTRACT
BACKGROUND: The aim of the present study was to evaluate the role of interleukin (IL)-6-634 polymorphism in neonatal disorders such as bronchopulmonary dysplasia (BPD) and periventricular leukomalacia (PVL) in very low-birthweight (VLBW) infants. METHODS: This prospective cohort study included 202 infants (gestational age at birth, 23-34 weeks; birthweight, 500-1499 g). Genotypic analysis (polymerase chain reaction-restriction fragment length polymorphism) was performed with DNA extracted from whole-blood samples. RESULTS: Genotype distribution (66.8% CC, 28.2% CG, 5.0% GG) was similar to that in the adult Japanese population. BPD occurred in 85 infants (42.1%) among 202 VLBW infants. The duration of O(2) therapy in infants with CG/GG genotypes was significantly longer than that in infants with the CC genotype (CG/GG vs CC: 40.3 ± 52.2 days vs 28.4 ± 32.6 days, P < 0.05), but the prevalence of BPD was not associated with the CG/GG genotype (CG/GG, 40.0%; CC, 46.3%, P= 0.24). Infants with CG/GG genotypes were more likely to have received postnatal corticosteroid therapy for BPD than those with the CC genotype (CG/GG vs CC: 20.9% vs 11.1%, P = 0.05). PVL occurred in six infants (3.0%). There was no significant difference in the prevalence of PVL among IL-6-634 polymorphisms (CG/GG, 3.0%; CC, 3.0%, P = 0.65). CONCLUSIONS: IL-6-634 polymorphism is associated with duration of oxygen therapy in VLBW infants. This suggests that the IL-6-634 polymorphism G allele is an aggravating factor of BPD. IL-6-634 polymorphism is not associated with PVL.
Subject(s)
Bronchopulmonary Dysplasia/genetics , Interleukin-6/genetics , Leukomalacia, Periventricular/genetics , Polymorphism, Single Nucleotide , Cohort Studies , Female , Humans , Infant, Newborn , Infant, Very Low Birth Weight , Male , Prospective StudiesABSTRACT
Morgagni hernia is an extremely rare form of congenital diaphragmatic hernia. Only 6 cases of this condition have been reported in the English literature, as diagnosed prenatally. The prognosis of the disease is determined by the severity of the pulmonary hypoplasia and associated anomalies. Here we report a case of Morgagni hernia with massive pericardial effusion diagnosed by ultrasonography and MRI during the second trimester, enabling planning of appropriate treatment in the pre- and perinatal periods.