gakkaizashi


THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.123, No.12, December 2019


Original Article
1. Renal Function of VLBW Infants at 9 Years of Age and the Results of Detailed Examination
2. A Questionnaire Survey on the Current Life Situation and Cognitive Impairment in School-Aged Children with Acquired Brain Injury
3. Erroneously Elevated Free Thyroxine Levels in a "One-step" Assay in Children with Down Syndrome
4. Clinical Features of Community-acquired Escherichia coli Bacteremia in Children
Case Report
1. Kawasaki Disease Complicated with Duodenal Perforation
2. A Boy Who Succumbed to Acute Cerebellitis with Rapidly Progressive Cerebellar Swelling: A Case Report
3. Sex-discordant Monochorionic Twins with Unbalanced Translocation
4. A Diabetic Pediatric Patient with Severe Motor and Intellectual Disabilities Was Treated with Flash Glucose Monitoring
5. A Case of Fatty Liver of Malnutrition due to Decreased Lactation Secretion in the Mother
Brief Report
Rate of Smoking among Mothers at a Pediatric Clinic


Original Article
Title
Renal Function of VLBW Infants at 9 Years of Age and the Results of Detailed Examination
Author
Reiko Kushima1) and Tae Omori2)
1)Department of Neonatology, Tokyo Metropolitan Bokutoh Hospital
2)Department of Pediatrics, Tokyo Metropolitan Bokutoh Hospital
Abstract
We assessed the renal function of about 300 very-low-birth-weight (VLBW) infants at 9 years of age. Serum creatinine (Cr) and cystatin C (CysC) were measured, and estimated glomerular filtration rate (eGFR) was calculated. Both Cr-eGFR and CysC-eGFR were lower as the birth weight (BW) was smaller. Cr-eGFR and CysC-eGFR of less than 90 mL/min/1.73 m2 were confirmed in 17% and 2% of VLBW infants, respectively. The smaller the BW, the higher the abnormality rate. Both Cr and CysC-eGFR were lower in small-for-gestational-age infants than appropriate-for-gestational-age infants.
Among the 14 patients who underwent detailed examination, about half had small kidneys on echography, and renal hypodysplasia was suspected.
In VLBW infants, it seemed necessary to check their renal function regularly and for a long period of time.




Original Article
Title
A Questionnaire Survey on the Current Life Situation and Cognitive Impairment in School-Aged Children with Acquired Brain Injury
Author
Ichiro Kuki Hiroshi Arai Risa Uno Mitsuru Kashiwagi Shuichi Shimakawa Tetsuzo Tagawa Takuya Tanabe Yasuhisa Toribe Toshisaburo Nagai and Yukiko Mogami
Osaka Pediatric Association, Coordination Committee of Hospitals and Clinics, Study Committee on Issues of Children with Disabilities
Abstract
Acquired brain injury (ABI) is commonly associated with severe cognitive impairment. However, there are few studies of school-aged children with mild cognitive impairment and the needs of these patients and their families. We conducted a questionnaire survey of patient characteristics, current life situation, and daily living outcomes of elementary/middle/high school students with ABI.
We obtained responses from 38 participants (79% response rate). The causes of injury included encephalitis/encephalopathy (n = 18) and traumatic brain injury (n = 11). Fourteen patients had epilepsy. Concerning school life, 23 (60%) patients were enrolled in mainstream classes, 28 (74%) reported difficulties in school and daily life, and 26 (68%) requested learning modifications and supports. Among the 31 respondents of the daily living survey, impaired visual perception was the most frequently reported complaint (n = 19; 61%). A correlational analysis of patients' current life situation showed a relationship between impaired visual perception and reduced ability to exercise (p < 0.001) and between memory impairment and epilepsy status (p = 0.009).
Children who sustained an ABI, even if the impairment was mild, demonstrated difficulties in school and daily life. Therefore, it is important to improve patient support systems and provide reasonable accommodations where required.




Original Article
Title
Erroneously Elevated Free Thyroxine Levels in a "One-step" Assay in Children with Down Syndrome
Author
Konomi Suzuki Tatsuya Fukasawa Tetsuo Hattori Tetsuo Kubota Yuichi Kato and Yuji Miyajima
Department of Pediatrics, Anjo Kosei Hospital
Abstract
Although thyroid function is measured using a "one-step" assay in many facilities, such an assay may yield erroneous results due to assay interference. However, assay interference can be avoided using a "two-step" assay.
Since thyroid disease is a common manifestation of Down syndrome (DS), it is recommended that thyroid function tests be routinely conducted for children with DS.
Between 2014 and 2015, thyroid function was tested in 1,292 children aged <15 years at our hospital using a one-step assay, including 71 children with DS and 1,221 without DS (non-DS). A retrospective analysis revealed elevated free thyroxine (FT3) levels (FT3 ≥ 7 pg/mL) with a nondecreased thyroid-stimulating hormone (TSH) level (TSH ≥ 0.5 μU/mL) in nine DS and three non-DS children. The two-step assay was used in five DS and two non-DS children to remeasure thyroid function. The results revealed normal FT3 levels in five DS and one non-DS children.
The number of children who showed elevated FT3 levels (FT3 ≥ 7 pg/mL) with a nondecreased TSH level (TSH ≥ 0.5 μU/mL) in the one-step assay was significantly greater in the DS group than in the non-DS group.
The number of children who showed normal FT3 levels in the two-step assay was greater in the DS group than in the non-DS group. Furthermore, children who showed normal FT3 levels in the two-step assay did not have any sign of thyrotoxic symptoms.
We consider it especially important to remeasure thyroid function using the two-step assay for children with DS who are asymptomatic for thyrotoxicosis and show one-step assay results indicating elevated FT3 and nondecreased TSH levels.




Original Article
Title
Clinical Features of Community-acquired Escherichia coli Bacteremia in Children
Author
Kyosuke Ibi Katsuhiko Kitazawa Mariko Arakawa Yuta Asakura Akihiro Iguchi Hironobu Kobayashi Masayoshi Senda and Akihito Honda
Department of Pediatrics, Asahi General Hospital
Abstract
Objective: The aim of this study was to assess the incidence and clinical characteristics of community-acquired Escherichia coli (E. coli) bacteremia in children. Methods: This study was a retrospective review of 31 children aged ≤15 years diagnosed with community-acquired E. coli bacteremia who were admitted to our hospital between January 1989 and December 2018. To analyze its incidence, we divided the study period into three 10-year periods. Clinical data included demographic features, diagnoses, antimicrobial susceptibilities of isolates, and outcomes. Results: The number and incidence (per 1,000 admissions) in the first, second, and third 10-year study periods were 8 (0.80), 7 (0.45), and 16 (1.24), respectively. A substantial increase in incidence was observed during the last 10 years. There were 17 cases of E. coli bacteremia (55%) in children aged ≥3 months. Among 23 children (74%) who had bacteremic urinary tract infections (UTI), vesicoureteral reflux (VUR) was found in 8 (35%) children, all of whom had VUR grade IV or V. In 8 non-UTI children, bacteremic episodes were associated with a wide variety of chronic or acute illnesses. During the last 10-year period, 6 of 16 E. coli isolates (38%) were found to be resistant to ampicillin, of which 2 were extended-spectrum β-lactamase-producing strains. One child with cardiopulmonary arrest died on hospital arrival. Conclusions: This study showed that the incidence of childhood community-acquired E. coli bacteremia increased over time and more than half of cases occurred in children aged ≥3 months. Although high-grade VUR appeared to be a contributing factor in children with UTI, accounting for 74% of all bacteremia cases, various comorbidities were observed in non-UTI children.




Case Report
Title
Kawasaki Disease Complicated with Duodenal Perforation
Author
Keisuke Oyatani1) Tomohiro Nawa1) Madoka Sawada1) Motoki Takamuro1) and Shigeto Fuse2)
1)Department of Pediatric Cardiology, Hokkaido Medical Center for Child Health and Rehabilitation
2)Department of Pediatrics, NTT Sapporo Medical Center
Abstract
Kawasaki disease (KD) is rarely associated with perforation of the gastrointestinal tract, a condition with a high risk of coronary artery aneurysms. To prevent sequelae, both surgical intervention and KD treatment must be applied without delay. A 1-year-old boy with no medical history was referred to a local hospital with a chief complaint of fever and systemic erythema multiforme on the second disease day. He was diagnosed with incomplete KD due to swelling of the hands, feet, and lymph glands in the neck and enlargement of the right coronary artery on cardiac ultrasonography. On hospitalization, intravenous immunoglobulin (IVIG) treatment was started, but he vomited 30 min later and developed abdominal distension and disordered consciousness 6 h later. The administration was therefore discontinued 7 h after initiation because of the possibility of adverse effects of IVIG. On the third disease day, an abdominal radiograph revealed free air, so the patient was transported to our hospital, and emergency surgery was performed. A diagnosis of duodenal perforation was made based on intraoperative findings. Although postoperative intensive care was necessary, it was considered that interruption of KD treatment should be avoided, so IVIG was resumed from the fourth disease day, and his fever was relieved on the fifth disease day. On the seventh disease day, oral aspirin was started with the permission of the surgeon. He was discharged on the 27th disease day without apparent sequelae to the digestive tract or coronary arteries.




Case Report
Title
A Boy Who Succumbed to Acute Cerebellitis with Rapidly Progressive Cerebellar Swelling: A Case Report
Author
Norihito Ikenobe1) Atsumi Hanawa1) Ryuta Tanaka1)2) Fujiko Fukushima1) Reiko Kyodo1) Tatsuo Kono3)4) Yukitoshi Takahashi5) and Isho Izumi1)
1)Department of Pediatrics, Ibaraki Children's Hospital
2)Department of Child Health, Faculty of Medicine, University of Tsukuba
3)Department of Radiology, Ibaraki Children's Hospital
4)Department of Radiology, Tokyo Metropolitan Children's Medical Center
5)Department of Pediatrics, National Epilepsy Center, Shizuoka Institute of Epilepsy and Neurological Disorders
Abstract
<Introduction> As acute cerebellitis is a rare but sporadically lethal disorder in children, early diagnosis and preparation against rapid aggravation are imperative. <Case report> A 12-year-old boy was admitted to our hospital because of dysarthria, ataxic gait (day 1), and convulsion (day 2) after an upper respiratory infection. Despite the standard MRI results on days 2 and 3, consciousness disturbance was extended. Day 5 MRI demonstrated a cerebellar lesion, confirming the diagnosis of acute cerebellitis. Steroid pulse therapy was initiated immediately; however, he displayed remarkable instability of the heart rate and slipped into a coma and respiratory arrest on day 7. Brain MRI revealed brainstem compression, obstructive hydrocephalus, and transforaminal herniation due to the progressive cerebellar swelling. The patient was clinically judged as brain dead, and he died on day 30. High serum antibody titers and a positive LAMP assay result from the throat indicated the presence of Mycoplasma pneumoniae infection. We also detected antibodies against glutamate receptor in the cerebrospinal fluid. <Discussion and conclusions> Inflammatory response due to M. pneumoniae infection could be involved in the pathophysiology and neurological symptoms. This report recommends reassessment by brain MRI for patients exhibiting prolonged unconsciousness and immediate consultation with a neurosurgeon after the appearance of cerebellar swelling.




Case Report
Title
Sex-discordant Monochorionic Twins with Unbalanced Translocation
Author
Yohei Kume1)2) Hiromichi Ariga1) Takashi Ono1) Kazufumi Yaginuma1) Yasuko Shibukawa1) Yutaka Fukuda1) Tomoo Fujiki1) and Katsutoshi Nagasawa1)
1)Department of Pediatrics, Takeda General Hospital
2)Department of Pediatrics, Fukushima Medical University
Abstract
The majority of monochorionic twins are monozygous; however, opposite-sex twins may be born when fertilized embryos are mosaic, especially 45,X/46,XY mosaic, according to previous reports. Here, we report a case of monochorionic diamniotic twins identified in early pregnancy by obstetric ultrasound. Twin I, whose karyotype was 45,X as assessed by amniotic fluid chromosomal test, developed cystic hygroma at 15 weeks of gestation and died at 24 weeks.
In twin II, chromosome analysis after birth revealed a karyotype of mos 46,X,der(Y)t(Y; 12)[8]/45,X[2], an emerged partial addition of 12q24.23-33 to Y chromosome, and a partial deletion of Yq11.223-qter by array comparative genomic hybridization (aCGH). Our case was natural pregnancy with one sac, and the twins shared a 45,X karyotype. Therefore, we considered them as monochorionic twins of opposite sex due to mosaic embryos.
Twin II with analogous symptoms of 12q trisomy also has mild mental retardation, right hydronephrosis, umbilical hernia, and skin hemangioma. He is currently 2 years 9 months old and receives rehabilitation. Due to a sex chromosome mosaic and a partial deletion of Y chromosome, it is necessary to follow up the patient for complications such as abnormal spermatogenesis.
In monochorionic twins, even though it is rare, we should recognize the possibility of discordant sex. Furthermore, if one infant shows Turner syndrome, the other infant may have chromosomal abnormalities such as sex chromosome mosaics.




Case Report
Title
A Diabetic Pediatric Patient with Severe Motor and Intellectual Disabilities Was Treated with Flash Glucose Monitoring
Author
Shunsuke Shimazaki1) Itsuro Kazukawa1) Kyoko Mori1) Makiko Kihara1) Masanori Minagawa1) Akiko Yamaide2) Minako Tomiita2) and Akira Hoshioka2)
1)Department of Endocrinology, Chiba Children's Hospital
2)Department of Allergy and Rheumatology, Chiba Children's Hospital
Abstract
A 10-year-old girl with severe motor and intellectual disabilities who received a nonlipid formula (Peptino®) via enteral feeding treatment for allergic enteritis was presented to our hospital with hypothermia, and she was diagnosed with type 2 diabetes. She was initially treated with continuous intravenous insulin therapy. This was followed by multiple daily injection (MDI) therapy, after which she was discharged from the hospital. However, hyperglycemia remained uncontrolled due to enteral feeding. Therefore, MDI therapy was replaced by continuous subcutaneous insulin infusion (CSII) therapy and flash glucose monitoring (FGM), which resulted in better glycemic control.
It is well known that hyperglycemia is one of the complications of tube feeding. In this case, a high-carbohydrate and low-lipid formula was believed to have induced the onset of diabetes. The stress of medical care was reduced by the use of CSII therapy. Better glycemic control was achieved with FGM because hyperglycemia caused by the tube feeding could be resolved.




Case Report
Title
A Case of Fatty Liver of Malnutrition due to Decreased Lactation Secretion in the Mother
Author
Asumi Yoshida1) Shingo Ishimori1)2) Ryosuke Bo3) Miho Sakata1) Hiroyuki Awano3) Yoshinobu Oyazato1) Atsushi Nishiyama1) and Masahiko Yonetani1)
1)Department of Pediatrics, Kakogawa Central City Hospital
2)Department of Pediatrics, Takatsuki General Hospital
3)Department of Pediatrics, Kobe University Graduate School of Medicine
Abstract
A girl was delivered at 38 weeks' gestation, and her mother had a normal pregnancy. She grew with normal exercise and mental development until 8 months old. She then considerably regressed as shown by neither laughing nor pulling up to standing at 10 months old. She had severe weight loss because of the decrease in the secretion of lactation of her mother. A laboratory examination showed increased liver enzymes, hypoglycemia, and an elevated blood lactic acid level. Ultrasonography showed an enlarged liver, and her blood examination did not indicate viral hepatitis or a congenital metabolic disorder. Improvement of her nutritional condition with tube feeding of milk returned the liver enzymes and lactic acid levels and enlarged liver to normal. She was finally diagnosed with fatty liver of malnutrition due to a small amount of lactation. Malnutrition is a rare cause of fatty liver in infants in developed countries. However, poor lactation of the mother should be considered as the cause of fatty liver enlargement to distinguish it from congenital metabolic disorders.




Brief Report
Title
Rate of Smoking among Mothers at a Pediatric Clinic
Author
Ayumi Murakoso-Matsumoto1) Mai Itai2) and Akiko Sasaki2)
1)Treponte Children's Clinic
2)Tokyo Medical and Dental University, Graduates School of Health Care Sciences
Abstract
We investigated the smoking rates among mothers in a pediatric clinic. An anonymous self-administered questionnaire survey was conducted with 688 mothers of pediatric patients who had visited our clinic from March to May 2018. The number of mothers who had smoked before pregnancy was 133. Of these, 115 (86.5%) had quit smoking when they became pregnant. After childbirth, 28.7% of these mothers had resumed smoking. The resmoking rate of mothers with children aged >6 years was significantly higher than that of mothers with children aged <5 years. We are concerned that mothers who continued smoking during pregnancy might have little interest in their children's health; thus, we believe that there is a cause for concern regarding a nurturing environment for the child and a healthy mother-child relationship. We consider that continuous support by pediatricians might be effective because one of the reasons for resmoking seems to be decreased postdelivery motivation.




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