Swimming was associated with rhinitis and pollinosis in a duration-dependent manner.

BACKGROUND: Previous studies have reported conflicting results regarding the effects of childhood swimming on respiratory allergic symptoms. We investigated the relationship between swimming and respiratory allergic symptoms in schoolchildren. METHODS: A questionnaire regarding participation in sports club activities and respiratory allergic symptoms in schoolchildren was distributed to the parents of all 6853 public school students (aged 6-14 years) in Omihachiman City, Shiga, Japan. The relationships between participation in sports club activities and the prevalence of respiratory allergic symptoms were analyzed by multivariable logistic regression analyses. RESULTS: Questionnaires were returned for 4991 schoolchildren (response rate: 72.8%). Logistic regression analysis revealed significant positive associations between swimming and rhinitis (42.9% vs. 38.9%; adjusted odds ratio, 1.26; 95% confidence interval, 1.10-1.44), and swimming and pollinosis (32.1% vs. 28.1%; adjusted odds ratio, 1.28; 95% confidence interval, 1.11-1.47). The duration of participation in swimming activities was also significantly positively associated with the prevalence of rhinitis and pollinosis. Those who had participated in swimming activities for 6 years or more showed significantly higher prevalences of rhinitis and pollinosis (46.3% and 36.4%, respectively) than those without swimming activities (38.9% and 28.1%, respectively) and those who had participated in swimming activities for 5 years or less (40.1% and 28.5%, respectively). CONCLUSIONS: Swimming was associated with the prevalence of rhinitis and pollinosis in schoolchildren, especially among those who had participated in swimming activities for 6 years or more. Preventive measures and early interventions for rhinitis and pollinosis should be recommended to these children.

Higher Intake of Nutrients with Antioxidant and Anti-Inflammatory Effects among Children with Cow's Milk Allergy According to the Degree of Elimination.

Children with cow's milk allergy (CMA) have lower bone mineral density and shorter height than children without CMA or non-food allergy (FA), and their final height is also affected. Appropriate nutritional guidance for CMA children is therefore needed. The purpose of this study was to conduct a dietary survey of CMA children and to compare their characteristics according to the degree of elimination. A dietary survey of FA children attending the allergy clinic at Shiga Medical Center for Children, Shiga, Japan, was conducted using the brief-type self-administered diet history questionnaire for Japanese children and adolescents (BDHQ15y). To objectively assess the intake of fruit and vegetables, skin carotenoid levels were measured using the Veggie Meter®. Regarding the degree of CM elimination, of the 147 FA children (89 boys, 58 girls) recruited, 38, 27, and 82 children were allocated to complete-, partial-, and non-elimination groups, respectively. In analyzing the data, boys and girls were combined because there was no difference in sex ratio among the groups. Significantly lower intakes of calcium, animal proteins, and vitamin B2 were observed with increasing degree of CM elimination. Conversely, significantly greater intakes of vegetable proteins, ß carotene, α tocopherol, vitamin C, and polyunsaturated fatty acids, as well as significantly higher levels of skin carotenoids, were observed with increasing degree of CM elimination. Intakes of many nutrients with antioxidant and anti-inflammatory effects were higher with increasing degree of CM elimination, presumably reflecting the efforts of caregivers in identifying alternative foods. However, calcium intake was still inadequate, indicating a need for further encouragement of calcium intake in CMA children.

Association of Skin Carotenoid Score and Food Intake among School Children: A Multicenter Cross-Sectional Study.

This study aimed to evaluate the associations between skin carotenoid measurements and fruit and vegetables intake among school children. We conducted a cross-sectional study for 10-y-old children in 2020 in Japan. We compared the optical skin carotenoid score, measured using Veggie Meter®, and food intake, based on a self-administered questionnaire. Among the 328 children who were registered in the three primary schools, 321 (97.9%) agreed to undergo skin carotenoid measurements. None of the children were unable to undergo the examination or experienced any adverse events. Among the 321 children who underwent skin carotenoid measurements, 315 (96.0%) responded to the questionnaire. The baseline characteristics showed that 160 (50.8%) children were boys. The median (interquartile range) skin carotenoid score was 335 (277-407) points. The minimum and maximum scores were 138 and 822 points, respectively, and the mean score (standard deviation) was 349 (104) points. The multivariable model showed a higher intake of fruits and green-yellow vegetables and more exercises were independently associated with higher skin carotenoid scores. We showed positive associations between higher skin carotenoid scores and higher intake of fruits and green-yellow vegetables and more exercise. The skin carotenoid score could be used to promote better food intake among children.

Helicobacter cinaedi-Associated Refractory Cellulitis in Patients with X-Linked Agammaglobulinemia.

X-linked agammaglobulinemia (XLA) is characterized by severe or recurrent infections, hypogammaglobulinemia, and circulating B cell deficiency. The frequent pathogens seen in patients with XLA include Streptococcus pneumoniae, Haemophilus influenzae, Pseudomonas aeruginosa, and enterovirus as well as Campylobacter and Helicobacter species. Here, we describe two patients with XLA who developed cellulitis and bacteremia caused by Helicobacter cinaedi even when administered an appropriate immunoglobulin replacement therapy. H. cinaedi may be difficult to isolate using a conventional blood culture system and could be identified by sequence analysis and mass spectrometry. H. cinaedi infection causes recurrent symptoms frequently, and patients require a long course of antibiotic treatment. Recently, the case of non-H. pylori Helicobacter (NHPH) infection such as H. cinaedi and H. bilis infection is increasing in number in patients with XLA. Systemic NHPH infection should be suspected, and extensive microbiological analysis should be performed to appropriately treat patients with XLA who present with fever and skin lesions.

Negative Association in Reported Dietary Energy Intake and Physique in Japanese Schoolchildren.

Appropriate dietary assessment and health education are necessary for children to achieve a healthy physique. To explore the relationship between habitual reported dietary energy intake (EI) and physique in elementary schoolchildren by sex and age, we conducted a longitudinal study, in the fiscal year 2011, that included all elementary schools in Omihachiman City, Shiga Prefecture, Japan. The study lasted for four consecutive years, ending in fiscal year 2014, and included 545 7-y-old schoolchildren in the target city. The subjects completed a brief self-administered diet history questionnaire with their guardians. The results of the study demonstrated a negative relationship between energy intake and the estimated energy requirement ratio and body mass index percentile values for both 7-, 9-, and 10-y-old boys and 7- to 10-y-old girls. These results suggest that there is a need to keep in consideration the under-reporting of obese children and over-reporting of lean children for dietary energy evaluation.

Risk Factors for Cerebral Infarction in Duchenne Muscular Dystrophy: Review With our 2 Cases.

BACKGROUND: Although the incidence of cerebral infarction is higher in Duchenne muscular dystrophy (.75 per 100) than in the general population (7.5-11.4 per 100 000), only 18 cases have been reported, and prevention and management guidelines for infarction in this disorder remain lacking. PATIENTS AND METHODS: We encountered 2 cases of Duchenne muscular dystrophy with cerebral infarction. To clarify risk factors for such infarction in Duchenne muscular dystrophy, we reviewed 20 cases, including our 2 patients. RESULTS: Age at onset of infarction ranged from 4 to 31 years (n = 19). Most patients were 16-21 years old (14 of 19; 73.7%). Eighteen patients (90%) had dilated cardiomyopathy (DCM), showing a higher frequency than in the age-matched general Duchenne muscular dystrophy population. Left ventricular ejection fraction (LVEF) ranged from 10.2% to 42% (median, 20%; n = 9). Detectable cardiac thrombus and atrial fibrillation were rare (2 of 17; 11.8%, and 1 of 17; 5.9%, respectively). CONCLUSIONS: Presence of DCM with low LVEF seems to be the strongest risk factor for cerebral infarction in Duchenne muscular dystrophy.

Fruit intake reduces the onset of respiratory allergic symptoms in schoolchildren.

BACKGROUND: Previous studies have shown that dietary pattern is associated with allergy prevention. METHODS: We conducted a prospective cohort study on all primary schools in Omihachiman City, Shiga Prefecture, Japan. Questionnaires regarding allergic symptoms and diet were distributed to the parents of all 759 7-year-old schoolchildren for 4 consecutive years, from 2011 to 2014. Specific immunoglobulin E to inhalant allergens was measured at 10 years of age. Participants were then categorized as low, medium, or high intake during the study period for four food groups (fruits, vegetables, fish, and beans). Logistic regression analysis was performed to estimate odds ratios and 95% confidence intervals. RESULTS: A total of 520 children (68.5%) whose parents responded to the questionnaires all 4 years were included in the analysis. The prevalence of asthma, rhinitis, and any allergic symptoms at age 10 was significantly decreased with increases in fruit intake. In addition, the onset of any allergic symptoms during the study period was significantly decreased with increases in fruit intake (33.3%, 28.3%, and 14.3% in children with low, medium, and high fruit intake, respectively; P for trend =.01). The sensitization rate to ragweed at age 10 was significantly decreased with increases in fruit intake (P for trend =.046). No significant effect was observed for the other three food groups, except for the association between fish intake and new-onset asthma symptoms. CONCLUSIONS: These findings suggest that higher intake of fruit can help prevent respiratory allergic symptoms in schoolchildren.

Left ventricular noncompaction cardiomyopathy in a patient with trisomy 13: A report and review of the literature.

Left ventricular noncompaction cardiomyopathy (LVNC) is characterized by prominent trabecular meshwork, and it is thought to result from arrest of the normal compaction process during embryogenesis. Patients with LVNC may be asymptomatic or have symptoms ranging from heart failure to stroke, life-threatening arrhythmias, or sudden death. The frequency of LVNC in children has increased with longer clinical courses. About 80% of patients with trisomy 13 have a congenital cardiac abnormality, but a clinical description of LVNC with trisomy 13 is lacking because of its poor prognosis and lack of awareness about LVNC. We described a patient with trisomy 13 who was diagnosed with LVNC-dilated phenotype and died suddenly, as well as two additional patients with LVNC. All three patients had chronic heart failure without congenital heart disease and were treated with diuretics. To manage trisomy 13 with or without congenital heart disease, cardiac disease such as LVNC may present at any ages, and therefore cardiac evaluation should be considered as a part of their appropriate management.

Organizing pneumonia as an extraintestinal manifestation of Crohn's disease in a child.

Crohn's disease (CD) is a chronic inflammatory disorder with an unknown etiology that commonly involves the gastrointestinal tract, and bronchopulmonary manifestations only occur in 0.4% of cases. There have not been any reports about pulmonary involvement in pediatric CD patients. We experienced a 14-year-old boy with Crohn's disease diagnosed with organizing pneumonia by chest CT-guided biopsy examination. His pneumonia was intractable despite the administration of multiple antibiotics, and steroid therapy was very effective. In pediatric patients with CD whose lung disease does not respond to antibiotics, OP should be considered as a possible diagnosis.

ALDH18A1-related cutis laxa syndrome with cyclic vomiting.

Cutis laxa (CL) syndromes are connective tissue disorders characterized by redundant, sagging, inelastic and wrinkled skin, with organ involvement. Here, we describe a patient with ALDH18A1-related CL who developed cyclic vomiting. The patient was a 12-year-old boy who presented with poor postnatal growth, hypotonia, short stature, joint hyperlaxity, microcephaly, strabismus, bilateral cataracts, facial dysmorphism and severe mental retardation. Bone radiographs showed osteopenia and osteoporosis, and magnetic resonance angiography showed marked kinking and tortuosity of the brain vessels. These findings were clinically compatible with ALDH18A1-related CL. Molecular analysis revealed a de novo heterozygous mutation (p.R138Q) in ALDH18A1. No mutations were found in PYCR1 gene. The patient developed cyclic vomiting with decreased blood levels of ornithine, citrulline, arginine and proline without hyperammonemia and other hypoaminoacidemias were also found. ALDH18A1 encodes Δ(1)-pyrroline-5-carboxylate synthase, which is related to the biosynthesis of ornithine, citrulline, arginine, and proline. Cyclic vomiting has never been reported in other ALDH18A1-related CL patients. This is the first case report of ALDH18A1-related CL with cyclic vomiting associated with amino acid abnormalities.

Sports activities enhance the prevalence of rhinitis symptoms in schoolchildren.

BACKGROUND: To evaluate the association between sports activities and allergic symptoms, especially rhinitis, among schoolchildren. METHODS: This longitudinal survey of schoolchildren collected data from questionnaires regarding allergic symptoms based on the International Study of Asthma and Allergies in Childhood (ISAAC) program and sports participation that were distributed to the parents of children at all 12 public primary schools in Ohmi-Hachiman City, Shiga Prefecture, Japan. Data were collected annually from 2011 until 2014, when the children reached 10 years of age. Blood samples were obtained in 2014, and the levels of immunoglobulin (Ig)E specific to four inhalant allergens were measured. RESULTS: Data from 558 children were analyzed. At 10 years of age, prevalence of asthma and eczema did not differ significantly, while rhinitis was significantly higher (p = 0.009) among children who participated in sports. Prevalence of rhinitis increased as the frequency or duration of sports participation increased (p < 0.01). The prevalence of new-onset rhinitis increased significantly among 10-year-olds with increasing duration of participation in sports (p = 0.03). Among those who participated in continuous sports activities, the prevalence of rhinitis was significantly higher with prolonged eczema (p = 0.006). Sports activities did not increase sensitization to inhalant allergens. CONCLUSION: Sports activities enhance the prevalence of rhinitis in schoolchildren. Prolonged eczema, together with sports participation, further promotes the symptoms. The mechanisms of these novel findings warrant further investigation.

Severe acute abdomen caused by symptomatic Meckel's diverticulum in three children with trisomy 18.

Meckel's diverticulum (MD) is the most prevalent congenital anomaly of the gastrointestinal tract and often presents a diagnostic challenge. Patients with trisomy 18 frequently have MD, but the poor prognosis and lack of consensus regarding management for neonates has meant that precise information on the clinical manifestations in infants and children with MD is lacking. We describe the cases of three children with trisomy 18 who developed symptomatic MD. Intussusception was diagnosed in Patient 1, intestinal volvulus in Patient 2, and gastrointestinal bleeding in Patient 3. All three patients underwent surgical treatment and only the Patient 1 died due to pulmonary hypertensive crisis. The other two patients experienced no further episodes of abdominal symptoms. In patients with trisomy 18, although consideration of postoperative complications and prognosis after surgical treatment is necessary, symptomatic MD should carry a high index of suspicion in patients presenting with acute abdomen.

Relationship between physique and food avoidance in infants: a study conducted in a community setting in Japan.

The relationship between food avoidance during infancy and the growth of Japanese infants in a community health setting has not been well evaluated. In order to assess the growth of infants who avoided either of the three major allergen foods in Japan, eggs, milk or wheat, we employed the results of 4 physical checkups recorded in maternity passbooks and administrated a questionnaire on allergic diseases, height and weight at birth to the guardians of 1,132 infants at the age of 3.5 years. Data was obtained from 890 subjects (78.6%) and 662 subjects (58.5%) who met the inclusion criteria were analyzed. The height, weight and body mass index percentile scores of each subject were calculated. Subjects who avoided either of the three foods at 3.5 years had lower weight percentile scores at 1.5 years, lower height and weight percentile scores at 3.5 years, and lower weight growth rates, compared with the subjects who did not avoid any of the three foods at 3.5 years (P=0.02, 0.03, 0.03, 0.01). The results suggested that there was a negative relationship between physique and food avoidance in infants, and that physical and nutritional assessments are important for food avoiders.

Fever of unknown origin as the initial manifestation of valproate-induced Fanconi syndrome.

BACKGROUND: Valproate-induced Fanconi syndrome is a rare adverse effect of valproate. Severely disabled patients who require tube feeding are reported to be susceptible to valproate-induced Fanconi syndrome. Although most patients with valproate-induced Fanconi syndrome are asymptomatic and detected incidentally with findings such as hypophosphatemia, hypouricemia, increased urinary ß2-microglobulin, and generalized hyperaminoaciduria, clinical symptoms such as bone fracture, fever, tachypnea, and edema have been reported. PATIENT DESCRIPTION: This 15-year-old, severely disabled, tube-fed, male patient with cytochrome oxidase deficiency had taken valproate for 3 years when he developed fever for 3 weeks. Hypophosphatemia, hypouricemia, hypokalemia, increased urinary ß2-microglobulin, and generalized hyperaminoaciduria, as well as hypocarnitinemia, were found, indicating that he had Fanconi syndrome. Valproate was the most likely cause of Fanconi syndrome in this patient. After discontinuation of valproate, the fever resolved immediately, and the laboratory findings normalized. CONCLUSION: Valproate-induced Fanconi syndrome should be considered when individuals taking valproate develop fever of unknown origin.

[Status of children with food allergy who were prescribed an adrenaline autoinjector (epipen)].

PURPOSE: Both to evaluate the characteristics of food allergic children who were prescribed an adrenaline autoinjector and to assess whether it was used appropriately. METHODS: The characteristics of food allergic children who were prescribed an adrenaline autoinjector were investigated. Among these children, those who experienced severe anaphylaxis due to inadvertent ingestion were analyzed, as was whether and how the autoinjector was used. RESULTS: An adrenaline autoinjector was prescribed to 139 food allergic children, most often for egg, followed by milk and wheat allergies. Concomitant bronchial asthma, atopic dermatitis, and food allergies of other causes were present in 49 (35.3%), 68 (48.9%), and 102 cases (73.4%), respectively. The most frequent organ involved in anaphylaxis was the skin (94.2%), followed by the respiratory (78.5%), digestive (28.1%), and circulatory (24.8%) organs. A total of 24 cases experienced severe anaphylaxis after the prescription; however, the autoinjector was used in only six (25%) of those cases. The reasons given for lack of use included fear of use, unavailability of the autoinjector, prior improvement with use of an oral antihistamine and immediate visit to a hospital emergency department in eight, five, three and one case, respectively. CONCLUSION: These results suggest that the autoinjector is often not used appropriately after prescription. Therefore, children and their caregivers require more effective guidance on proper adrenaline autoinjector use.

Pathological fracture and pyogenic osteomyelitis in a patient with type 2 Gaucher disease.

In Gaucher disease (GD), enzyme replacement therapy (ERT) results in the alleviation of hematological abnormalities and visceral infiltration as well as improvement in quality of life and life-span. However, several years may be required for skeletal manifestations, which are usually observed in type 1 and 3 GD, to respond to ERT. Infants with type 2 GD rarely present skeletal manifestations because most of these patients die within the first 2 years of life before they develop skeletal involvement. The use of ERT may prolong the lifespan of these patients and influence the natural history of the disease. The present study reports a new natural history of treated GD in which a 2-year and 7-month-old girl with type 2 GD who was receiving ERT developed valproate-induced Fanconi syndrome, pathological fractures, and pyogenic osteomyelitis. In conclusion, skeletal disease may occur in any type of GD, and Fanconi syndrome may lead to severe skeletal complications in patients with GD.

Low birth weight is associated with high waist-to-height ratio in Japanese elementary school girls.

In Japan, the rate of low birth weight infants has increased, due to an increase in the number of women who smoke or are lean. A recent study showed that low birth weight was associated with a high adult waist-to-height ratio in adult Japanese women, but little data is available concerning children. In this cross-sectional study with 568 subjects (276 boys and 292 girls), we examined the association between birth weight and waist-to-height ratio in 7- or 8-year-old Japanese children, all born at full term. The mothers of the subjects responded to a questionnaire about the weight of the children at birth, and physical data were collected from the results of measurements conducted at each school. We divided the subjects into two groups by the median of the birth weight (3,000 g) by sex to elucidate the effects of birth weight on the waist-to-height ratio. There were 119 boys and 164 girls and 157 boys and 128 girls in the < 3,000 g and ≥ 3,000 g birth weight groups, respectively. The Mann-Whitney U test was used to compare the physical conditions in the two birth weight categories. Height was significantly lower in the birth weight < 3,000 g group among both the boys (P < 0.001) and girls (P < 0.001). The waist-to-height ratio was significantly higher in the < 3,000 g group in girls (P = 0.004), but not in the boys. Our results suggest that intrauterine environmental insults might have an effect on children, depending on sex.