Effects of subcutaneous immunotherapy in allergic rhinitis children sensitive to dust mites.

BACKGROUND: Subcutaneous immunotherapy (SCIT) is now the only treatment that can modify the natural course of allergic rhinitis (AR). However, not all children with AR benefit from SCIT. OBJECTIVE: To evaluate the efficacy of SCIT in dust-mites-induced AR children and explore correlative factors predicting treatment response to SCIT. METHODS: 225 children aged 4-17 years old with AR were recruited from January 2016 to September 2019, and monitored at baseline, 4, 12, and 24 months after the start of SCIT treatment. The visual-analogue-score (VAS) was used to assess the clinical symptoms. Multivariate binary logistic regression analyses and receiver operating characteristic curves were used to explore correlative factors in predicting the efficacy of SCIT. RESULTS: The significant declines in VAS started after 4 months of SCIT and continued to improve throughout the study compared with baseline. An increase in children's age (OR=0.688, 95%CI: 0.479-0.988) and those with allergic history (OR=0.097, 95%CI: 0.009-1.095) were negatively associated with the risk of poor efficacy. Polysensitized children were more likely to suffer poor efficacy (OR=15.511 95%CI: 1.319-182.355). The clinical response at month 4 (r=0.707) and month 12 (r=0.925) was related to that at month 24. The area under the curve (AUC) for improvement at month 4 and month 12 was 0.746 and 0.860, respectively. CONCLUSION: Our study confirmed the clinical efficacy of SCIT in AR children. Children with younger age, negative allergic history, and multiple allergens may predict a worse efficacy. The onset of action and the clinical response to SCIT in the second year can be predicted as early as by month 4.

Mild Encephalitis/Encephalopathy with Reversible Splenial Lesion Associated with Scrub Typhus in a Child: A Case Report.

Background: Mild encephalitis/encephalopathy with a reversible splenial lesion (MERS) is a rare clinicoradiological syndrome characterized by transient mild encephalopathy and magnetic resonance imaging (MRI) findings of a reversible lesion in the splenium of the corpus callosum (SCC). Multiple causes have been proposed for the pathogenesis of MERS, with infection as the most pre-eminent. Case Presentation: We report the case of a 10-year-old girl with MERS due to scrub typhus. Her clinical manifestations of headache and drowsiness, together with lesions involving the SCC, as shown by MRI, and their complete resolution upon follow-up fulfilled the diagnosis of MERS. At the same time, the characteristic eschar of the skin and the positive Weil-Felix test result confirmed the existence of scrub typhus infection. Conclusions: To the best of our knowledge, we described the first pediatric case of MERS associated with scrub typhus. The case indicated that an MERS patient with fever should be considered as possibly having a scrub typhus infection. The characteristic black eschar of scrub typhus generally occurs after bite of mite that is important and useful to the doctor for making proper diagnosis.

Congenital Brucellosis: A Case Report.

Background: Brucellosis is the most common zoonotic infection worldwide, and is caused by bacterial genus Brucella. The disease is rarely transmitted through human-to-human transmission. Few cases have been reported about vertical transmission of human brucellosis. Herein, we reported a case of congenital brucellosis, with clear evidence of pathogen detected in mother's placental specimen. Case Presentation: A 34-day-old girl was admitted to the department of pulmonology with fever for 8 days. Three blood samples and one sample of cerebrospinal fluid were positive for Brucella melitensis. The diagnosis of brucellosis and B. melitensis meningitis were established, along with hyperbilirubinemia and liver dysfunction. Treatment of rifampicin (for 6 weeks) and meropenem (for 2 weeks) was administered. However, the disease relapsed within 18 days. Thereafter, a combination therapy of rifampicin and sulfamethoxazole/trimethiprim (SMZ/TMP) was administered for 8 weeks. The disease relapsed again in 42 days. For chronic brucellosis, three courses of combination therapy of rifampicin and SMZ/TMP was administered. The mother had fatigue and arthralgia for 2 weeks, fever and membrane rupture 1 day before the baby was born. B. melitensis DNA was detected in the mother's placental specimen by next-generation sequencing and bacterial identification under microscope proved chorioamnionitis. Conclusions: We reported a confirmed case of congenital brucellosis. This disease should be closely monitored even in nonepidemic areas. The treatment of brucellosis in infancy faces challenges of drug choice and disease relapse.

Mitochondrial DNA insert into CD40 ligand gene-associated X-linked hyper-IgM syndrome.

BACKGROUND: X-linked hyper-IgM (X-HIGM), which results from mutations in the CD40LG gene located on chromosome Xq26.3, is the most common form of HIGM. To date, more than 130 variants of the CD40L gene have been reported. We described a patient with novel de novo nuclear mitochondrial DNA sequences (NUMTs) in the CD40LG gene that have resulted in X-HIGM. METHODS: Whole-exome sequencing (WES) analysis was used to screen for causal variants in the genome, and the candidate breakpoint was confirmed by Sanger sequencing. RESULTS: A new mutation of CD40LG, which deletes A at position 17 followed by a 147-nucleotide from mitochondrial DNA copies insertion in exon 1, was detected in a 20-month-old boy harbouring an X-HIGM combined with immunodeficiency syndrome. CONCLUSION: This is one of the few cases of a human genetic disease caused by nuclear mitochondrial DNA sequences (NUMTs). The presented data serve to demonstrate that de novo NUMT transfer of nucleic acid is a novel mechanism of X-HIGM.

A case report of pulmonary artery sling and situs inversus incompletes.

RATIONALE: Pulmonary artery sling (PAS) is a rare congenital anomaly. Associated airway anomalies and/or those of the cardiovascular system are present in about half the patients. Situs inversus is a rare disease in which organs of the chest and/or abdomen are arranged in a mirror image reversal of their normal position. Herein, we report a rare case of pulmonary artery sling and situs inversus incompletus, which has not yet been reported. PATIENT CONCERNS: A 10-year-old girl was admitted because of heart murmur for more than 9 years. On physical examination, the second heart sound was prominent, and a grade 2/6 systolic murmur was heard at the left mid-sternal border. Echocardiography revealed PAS and atrial septal defect (8.6 mm). A chest computer tomography angiograph demonstrated that she had lung inversus, right aortic arch, and right lung hypoplasia in addition to PAS, with a normal positioning of the heart. The PAS intersected and twisted across the bronchus, which was obviously narrowed. The PAS was type II B, since the carina was at the T6 level without a separate right upper lobe bronchus. DIAGNOSES: Her final diagnosis was that of PAS, tracheal stenosis, situs inversus incompletus, right lung hypoplasia, right aortic arch, ASD and PDA. INTERVENTIONS: She underwent one-stage total correction for her initial cardiovascular defects through median sternotomy under cardiopulmonary bypass support. OUTCOMES: She had an uneventful recovery and completely healthy following the procedure. LESSONS: A thorough examination before PAS surgery was essential in discovering and carefully evaluating complicated heart and lung anomalies.

Gastrodin Inhibits Virus Infection by Promoting the Production of Type I Interferon.

Type I interferon (IFN-I) plays a critical role in the antiviral immune response. However, viruses have developed different strategies to suppress the production of IFN-I for its own escape and amplification. Therefore, promoting the production of IFN-I is an effective strategy against virus infection. Gastrodin (GTD), a phenolic glucoside extracted from Gastrodia elata Blume, has been reported to play a protective role in some central nervous system -related diseases and is beneficial for the recovery of diseases by inhibiting inflammation. However, the effect of GTD on virus infection is largely unknown. Here we found GTD treatment increased the survival rate of mice infected with vesicular stomatitis virus (VSV) or herpes simplex virus-1 (HSV-1). The production of IFN-I was increased in GTD-treated mice or macrophages compared to the control group, during virus infection. Furthermore, the activation of interferon regulatory factor 3 (IRF3) was promoted by GTD in macrophages upon VSV and HSV-1 infection. Our results demonstrated that GTD could inhibit the VSV and HSV-1 infection by promoting the production of IFN-I in macrophages and might provide an effective strategy against virus infection.