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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.124, No.8, August 2020


Original Article
1. Trend of Appropriate Antibiotic Use at an Urgent Care Center for Children
2. Management of Childhood Chronic Functional Constipation in Primary Care
3. Present Status and Issues for Children with Medical Complexity in Tochigi Prefecture
Case Report
1. Congenital Combined Multiple Pituitary Hormone Deficiency Presenting Acute Respiratory and Hypoglycemia with Hyperammonemia
2. Three Cases of Takotsubo Cardiomyopathy Caused by Neurological Disease in Children
3. GQ1b Antibody-positive Miller Fisher Syndrome that Recurred Five Years after the Initial Presentation
4. Development of Nephrotic Syndrome during Bypass Therapy Accompanied by Inhibitor-positive Hemophilia B: A Case Report
5. Early Bordetella pertussis Re-infection or Recurrence in an Infant after Optimal Antibiotic Therapy
6. Anti-glomerular Basement Membrane Antibody Disease with Pulmonary Hemorrhage Alone and Seroconversion of Anti-glomerular Basement Membrane Antibodies 11 Months after Onset


Original Article
Title
Trend of Appropriate Antibiotic Use at an Urgent Care Center for Children
Author
Shogo Otake1)2) Yoshiki Kusama3) Hiroshi Okada1) Ken Tatsumi4) Masato Mori1) Ryugo Hiramoto1) and Masashi Kasai2)
1)Department of Pediatrics, Matsudo City General Hospital
2)Division of Infectious Disease, Hyogo Prefectural Kobe Children's Hospital
3)AMR Clinical Reference Center, Disease Control and Prevention Center, National Center for Global Health and Medicine
4)Tatsumi Kids Clinic
Abstract
It is important to evaluate appropriate antibiotic use at urgent care facilities, as many antibiotics are often prescribed. We evaluated antimicrobial prescription trends at an urgent care center for children in Matsudo to promote the appropriate use of antibiotics. We investigated the antibiotic prescriptions for 100 pediatric patients and the proportion of the type of antibiotics prescribed between 2015 and 2018. In addition, we surveyed the specialties of doctors who prescribed antibiotics and diagnoses for which antibiotics were prescribed in 2018. Between 2015 and 2018, the total number of antibiotics prescribed for 100 patients decreased from 8.03 to 5.08, and the proportion of prescribed amoxicillin increased from 56.8% to 73.7%. Pediatricians tended to prescribe amoxicillin more often than non-pediatricians, and non-pediatricians tended to prescribe cefdinir more often than pediatricians. Antibiotics were most often prescribed for group A streptococcal infections, for which 96.9% prescribed amoxicillin. Our study has suggested that factors promoting proper antibiotic use at our institution included restricting the selection of antibiotics, in addition to the proximity of Matsudo City General Hospital, which strongly promotes the proper use of antibiotics.




Original Article
Title
Management of Childhood Chronic Functional Constipation in Primary Care
Author
Kazuhiko Tomimoto
Tomimoto Children's Clinic
Abstract
【Background】
The cure rate of chronic functional constipation in children has been reported to range from 36% to 59% 12 months after treatment, but there are no reports in the field of primary care in Japan, and the critical point of maintenance therapy has not been clarified.
【Purpose and Method】
We examined factors related to the cure rate and treatment period in children with chronic functional constipation by comparing the conditions at 4 weeks prior to laxative discontinuation between the cure group and the relapse group.
【Result】
The cure rate after 12 months was 25.2%, whereas that for groups of children over 4 years old was 49.2%. Only the age at the time of the first visit was related to the duration of treatment. In comparison of the 133 cure cases with 106 relapse cases, the cure cases were related to the number of days of defecation, appropriate stool consistency, and complaint of abdominal pain. However, no relation was found between the rectal dilatation indicated by maximal rectal diameter in the past. Ensuring the number of days of defecation was significantly related to defecation training.
【Conclusion】
The short term prognosis of chronic functional constipation in childhood had a higher cure rate in the age group where defecation training is applicable.
In maintenance therapy, regular defecation is advised by using active laxative and regular defecation training that can keep soft stool close to type 5, because repeated painful defecation may lead to intractability. Children who complain of abdominal pain even with soft stools are relatively hard to cure and may be related to constipation-type irritable bowel syndrome.




Original Article
Title
Present Status and Issues for Children with Medical Complexity in Tochigi Prefecture
Author
Yuri Matsubara Yasuko Aoyama Koki Kosami Ryusuke Ae Nobuko Makino Shizukiyo Ishikawa and Yosikazu Nakamura
Department of Public Health, Jichi Medical University
Abstract
The number of children with medical complexity is increasing. In this study, data obtained from a survey conducted by Tochigi Prefecture was used for secondary analysis. The aim of this study was to reveal the needs of patients and suggest ways to enhance support.
The Division of Service of Tochigi Prefecture sent a questionnaire to the homes of children with medical complexity. The participants were children under 20 years old who receive public monetary support. This study was held from May 2017 to the end of June. We investigated the physical development, intellectual skills, and daily life of the participants, and analyzed the differences in the problems encountered by them.
The response rate was 54.3% and we estimated that almost 300 patients received medical care at home. The prevalence was 0.9 for a population under the age of 20 per 1,000 residents. Around 50% of respondents were under 6 years old, and 50% of patients were bedridden or non-ambulatory. However, 31% of patients had the ability to walk and run.
Both a lack of transportation service and economic support were the main issues faced by participants with artificial ventilators. In the case of tube feeding, the percentage of caregivers who sleep less than 5 hours was significantly higher.
The proportion of patients with medical complexity was slightly higher in Tochigi prefecture compared with other prefectures in Japan. Therefore, there is a great need to provide more appropriate services for each patient.




Case Report
Title
Congenital Combined Multiple Pituitary Hormone Deficiency Presenting Acute Respiratory and Hypoglycemia with Hyperammonemia
Author
Eriko Eto Tomoyo Itonaga Fumika Kawano-Matsuda Kazuhito Sekiguchi Miwako Maeda Tomoki Maeda and Kenji Ihara
Department of Pediatrics, Oita University Faculty of Medicine
Abstract
Hyperammonemia with acute circulatory failure and respiratory failure during the early neonatal period strongly suggests the presence of urea cycle disorders or organic acidemias. The early introduction of acute blood purification therapy in the neonatal intensive-care unit is required in such cases. However, congenital multiple pituitary hormone deficiency (MPHD) is sometimes complicated with hyperammonemia during the neonatal period. The early diagnosis of MPHD is difficult because of its nonspecific symptoms and very low incidence (about 1 per 100,000 births). We herein report a newborn boy presenting with apnea and hypoglycemia at eight hours after birth. He suffered from catecholamine-refractory hypotension, and hypoglycemia with marked hyperammonemia (183 μmol/L), suggesting the presence of congenital metabolic disorders. The clinical finding of a small penis indicated underlying hypogonadotropic hypogonadism, and an endocrinological examination supported a diagnosis of hypopituitarism at 20 h after birth. After introducing hormone replacement therapy, his respiratory and circulatory failure improved. Brain magnetic resonance imaging at 32 days of age showed pituitary hypoplasia and the absence of a posterior lobe on T1-weighted imaging. In summary, these results should remind all pediatricians and neonatologists that neonates with MPHD could demonstrate mild hyperammonemia early in the neonatal period.




Case Report
Title
Three Cases of Takotsubo Cardiomyopathy Caused by Neurological Disease in Children
Author
Ken Hayashi Toshikatsu Tanaka and Yasunobu Miki
Cardiology Department, Kobe Children's Hospital
Abstract
Takotsubo cardiomyopathy is a condition that often results from stress experienced by postmenopausal women. While there have been some reports of Takotsubo cardiomyopathy in children, it is considered to be rare and these cases are attributed to neurological disease. Between April 2017 and March 2019, we encountered 344 children in the pediatric intensive care unit at Kobe Children's Hospital; of these, 3 children were given a diagnosis of Takotsubo cardiomyopathy caused by neurological disease. Specifically, the causes were hydrocephalus, acute encephalopathy with biphasic seizures and late reduced diffusion, and subdural hematoma. All three children had systolic apical ballooning of the left ventricle, and 2 children also had pulmonary edema. We treated the underlying disease and used an inotropic agent to maintain brain perfusion pressure for a week. When we see neurological disease with circulatory failure and Takotsubo cardiomyopathy is strongly suspected, we should not perform invasive examination immediately and must carefully consider the use of inotropic agents, as they may result in iatrogenic worsening of cardiac function.




Case Report
Title
GQ1b Antibody-positive Miller Fisher Syndrome that Recurred Five Years after the Initial Presentation
Author
Kentaro Okada1) Yuichiro Muto1) Kuniyasu Wada2) Katsuki Hirai1) and Masahiro Migita1)
1)Department of Pediatrics, Japanese Red Cross Kumamoto Hospital
2)Department of Neurology, Japanese Red Cross Kumamoto Hospital
Abstract
Miller Fisher Syndrome is considered to be a variant of Guillain-Barré syndrome and is characterized by the acute onset of external ophthalmoplegia, ataxia, and areflexia. Miller Fisher Syndrome is usually a monophasic disease, and recurrence is extremely rare.
We herein report a boy who presented with recurrent Miller Fisher Syndrome that developed five years after the initial episode at nine years of age. During both the initial and recurrent episodes, he developed external ophthalmoplegia, ataxia, and areflexia after experiencing a previous infection. The clinical features of the recurrent episode were more aggressive than those of the initial one, with complete ophthalmoplegia and severe ataxia. In both episodes, the anti-GQ1b antibody was positive, and he recovered after administration of intravenous immunoglobulin therapy.
Miller Fisher Syndrome may recur during childhood, and symptoms may be more serious at the time of recurrence than they initially were.




Case Report
Title
Development of Nephrotic Syndrome during Bypass Therapy Accompanied by Inhibitor-positive Hemophilia B: A Case Report
Author
Chisaki Moriwaki1) Yasuko Okumura1) Yosuke Miyamoto1) Kohei Hayashi1) Toshihiko Imamura2) Keiji Nogami3) and Zenro Kizaki1)
1)Japanese Red Cross Kyoto Daiichi Hospital Pediatrics
2)Kyoto Prefectural University of Medicine Graduate School of Medicine Pediatrics
3)Department of Pediatrics, Nara Medical University
Abstract
Inhibitor development in hemophilia is a serious problem in the treatment of hemophilia. There are also some problems faced while treating patients with inhibitor-positive hemophilia B, one of which is nephrotic syndrome. A 2-year-old boy with hemophilia B developed inhibitor at the age of 9 months. He then received rFVIIa on demand until he developed hemorrhage of the left temporal lobe at the age of 1 year 5 months. Consequently, he received hemostatic regular replacement therapy of activated prothrombin complex concentrate (aPCC). However, after 10 months, he developed nephrotic syndrome, but he did not achieve remission after one month of steroid treatment or after steroid pulse therapy. Several studies have reported on nephrotic syndrome during immune tolerance induction, but to the best of our knowledge, there have been no studies on the occurrence of nephrotic syndrome during aPCC treatment. It is possible to develop nephrotic syndrome even during bypass therapy with aPCC, therefore, regular urinalysis should be conducted to monitor proteinuria.




Case Report
Title
Early Bordetella pertussis Re-infection or Recurrence in an Infant after Optimal Antibiotic Therapy
Author
Shigeki Ishii1) Ryohei Yokoyama1) Kazunari Takamura1) Noriko Hidaka1) Yoshiko Yamamura1) Tomoko Ohhira1) Hidemi Shimonodan1) Keigo Nakatani1) Yuka Mihara2) and Kenji Okada3)
1)Department of Pediatrics, Miyazaki Prefectural Miyazaki Hospital
2)Department of Pediatrics, Kariya Toyota General Hospital
3)Division of Basic Medical Science and Fundamental Nursing, Department of Nursing, Fukuoka Nursing College
Abstract
A 59-day-old boy experienced reinfection or recurrence of Bordetella pertussis infection 1 month after receiving optimal antibiotic therapy for pertussis. Five days prior to his first admission to our hospital, he developed a mild cough for which his family physician prescribed clarithromycin due to a history of close contact with his 7-year-old aunt in whom pertussis was previously diagnosed on polymerase chain reaction (PCR) testing. Despite treatment, the boy's paroxysmal cough gradually worsened, and he was admitted due to pertussis diagnosed on PCR. Seven days later, he was discharged without complications. Antibiotic post-exposure prophylaxis (PEP) should have been administered to the entire family to prevent a secondary infection, but antibiotics were only prescribed to a few family members.
Three weeks after discharge, the boy was re-admitted due to recurrence of paroxysmal cough. B. pertussis was isolated from a culture test performed at the time of re-admission. Based on these results, we concluded that he had B. pertussis reinfection or recurrence.
PEP for pertussis prevents secondary infections. However, it is unclear how often PEP is correctly administered in Japan. When treating pertussis patients, pediatricians should consider PEP for all close members of the household.




Case Report
Title
Anti-glomerular Basement Membrane Antibody Disease with Pulmonary Hemorrhage Alone and Seroconversion of Anti-glomerular Basement Membrane Antibodies 11 Months after Onset
Author
Kenji Bando Seiko Murakami and Tomo Sawada
Department of Pediatrics, Izumi City General Hospital
Abstract
Anti-glomerular basement membrane disease (AGBMD) is a rare immune complex-related small-vessel vasculitis caused by AGBM antibodies (Ab) and often presents with rapidly progressive glomerulonephritis with or without alveolar hemorrhage. AGBMD rarely presents with lung involvement alone and is often reported as AGBMD with normal renal function. Although serological tests, such as enzyme-linked immunosorbent assay (ELISA), are highly sensitive and useful, some patients with negative AGBM Ab are diagnosed by renal biopsy. This report examines a unique case of AGBMD with pulmonary hemorrhage alone and the occurrence of AGBM Ab seroconversion 11 months after the disease onset. An 11-year-old girl was admitted to our hospital with hemoptysis and anemia. She was initially given a diagnosis of idiopathic pulmonary hemosiderosis (IPH) based on negative results for AGBM Ab by chemiluminescent enzyme immunoassay (CLEIA). Her hemoptysis recurred several times despite corticosteroid treatment. Additionally, CLEIA performed 11 months after the disease onset was positive for AGBM Ab (13.9 U/mL) and therefore AGBMD was diagnosed. The addition of azathioprine prevented the deterioration of her symptoms, and she has maintained remission for 30 months with inhaled corticosteroids alone. Her renal function based on serum creatinine levels and urinalysis has been normal throughout the disease course. AGBM Ab are often undetectable in patients with AGBMD and pulmonary hemorrhage alone. The current case suggests that in some patients, IPH may be diagnosed and that AGBM Ab levels should be examined more than once to accurately understand the epidemiology and pathophysiology of AGBMD as well as IPH.




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