BackgroundAlthough late-onset circulatory collapse (LCC) is widely recognized in Japan, its etiology and the reason for center variation in its incidence remain unclear. This study’s objectives were to identify the perinatal and neonatal factors related to LCC and to estimate the factors related to the center variation in the incidence of LCC.MethodsExtremely preterm infants born between 2008 and 2012 who were registered in the database of the Neonatal Research Network, Japan were retrospectively analyzed. LCC was defined as a clinical diagnosis of LCC and the administration of steroids. We first identified the factors that were significantly related to LCC. We then examined the cause of the center variation in the incidence of LCC, using the standardized incidence ratios (SIRs) of LCC and individual factors.ResultsThe factors significantly associated with LCC included low gestational age (odds ratio [OR]: 1.13), small for date (OR: 1.43), male sex (OR: 1.26), antenatal steroid use (OR: 1.19), respiratory distress syndrome (OR: 1.25), chronic lung disease at 36 weeks (OR: 1.16), periventricular leukomalacia (PVL) (OR: 2.57), necrotizing enterocolitis (OR: 0.59), retinopathy of prematurity (ROP) (OR: 1.73), high-frequency oscillating ventilation (HFOV) use (OR: 1.31), parenteral nutrition (OR: 1.38), and red blood cell (RBC) transfusion (OR: 1.94). The SIR of LCC ranged from 0.05 to 2.94, and was positively correlated with SIRs of PVL, ROP, HFOV use and RBC transfusion.ConclusionPVL, ROP, HFOV use and RBC transfusion were found to be correlated with the center variation in the incidence of LCC.

We encountered a neonatal patient with hemophagocytic lymphohistiocytosis (HLH) whose mother was positive for anti-Ro/SSA and anti-La/SSB antibodies. Complete atrioventricular block was found in a male patient at 29 weeks of gestation. The patient was born at 40 weeks of gestation. He showed severe circulatory disturbance at 22 h after the birth, and he also had elevated serum levels of aspartate aminotransferase (1027 IU l(-1)), alanine aminotransferase (121 IU l(-1)), lactic dehydrogenase (3490 IU l(-1)), ferritin (9769.7 ng ml(-1)) and soluble interleukin-2 (IL-2) receptor (3230 U ml(-1)). We could not find any known HLH genetic abnormality in the patient, but he fulfilled seven of the eight criteria for HLH. Serum levels of IL-6 and IL-8 had been already elevated in his cord blood, and serum levels of granulocyte-macrophage colony-stimulating factor and IL-8 were significantly increased on the second day of life. His symptoms regressed with the administration of hydrocortisone. We presumed that transplacental transfer of maternal antibodies could be related to the occurrence of HLH.

This study assessed the psychometric properties of the Japanese version of the Eyberg Child Behavior Inventory (ECBI) in children in clinical and non‐clinical settings in Japan. Validation of the ECBI for clinical and non‐clinical participants (N = 128, 2–7 years of age) was evaluated. First, we evaluated the internal consistency reliability of the ECBI Problem and Intensity scales. We evaluated the construct and criterion‐referenced validity by comparing scores among the subscales of the ECBI, Child Behavior Checklist (CBCL), and Japanese versions of the Parenting Stress Index‐Short Form (PSI‐SF) and the Beck Depression Inventory‐II (BDI‐II). Results showed that Cronbach's alphas for both the Intensity and Problem scores were .91 and .92, respectively, which reflects high internal consistency. Results also showed that both the ECBI Intensity and Problem scores were significantly correlated with all subscales of the CBCL, PSI‐SF, and BDI‐II. These data suggest that the Japanese version of the ECBI is a psychometrically sound measure for assessing behavior problems in Japanese children.

We studied the cytokine profile of two siblings with neonatal lupus erythematosus (NLE) born to a mother positive for serum anti-Ro and -La antibodies, who did not receive any medication during the two pregnancies. The first sibling was found to have complete atrioventricular block in utero and became severely ill after birth. He fulfilled the diagnostic criteria for hemophagocytic lymphohistiocytosis on day 2. The second sibling did not have any fetal symptoms. He was generally stable after birth, but with typical skin rash. Laboratory data suggested that they both had hypercytokinemia during the neonatal period, requiring corticosteroid treatment. Interleukin (IL)-6, interferon-γ, IL-8 and monocyte chemotactic protein-1 were elevated in both cases, while IL-12, IL-13 and IL-17 were elevated only in the second sibling. Comparison of the cytokine profiles suggests the potential roles of different cytokines in the onset and clinical manifestations of NLE.

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