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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.123, No.7, July 2019


Original Article
1. Lung Ultrasonography for Neonatal Pneumothorax
2. The Need to Investigate Urological Abnormalities in Acute Epididymitis in Children
3. Effectiveness of Rotavirus Vaccines for Decrease of Rotavirus Gastroenteritis in a Pediatric Outpatient Clinic, Analyzed by Vesikari Score
Case Report
1. Three Cases of Percutaneous Closure for Congenital Portosystemic Shunt with Hypergalactosemia
2. A sibling case of nephrogenic diabetes insipidus with different developmental progress depending on the time of treatment intervention
3. A Comparison of Cytokines in a Child with Acute Focal Bacterial Nephritis with Mild Encephalopathy and Acute Pyelonephritis
4. A Case of 46, XX Disorders of Sex Development Due to Maternal Adrenal Adenoma
5. Acute Focal Bacterial Nephritis Caused by Staphylococcus aureus in a Boy with Atopic Dermatitis
6. Three Cases of Drug Intoxication False-negative on a Handy Drug Screening Kit
7. A Case of a Transfer from a Secondary or Tertiary Medical Institution to Home Care through a Home Care Support Hospital
Brief Report
Current Risk Assessment of Nicotine Poisoning from Ingestion of Heat-not-burn Tobacco: Eight Pediatric Cases


Original Article
Title
Lung Ultrasonography for Neonatal Pneumothorax
Author
Yusuke Hoshino Junichi Arai Yoshiya Yukitake Yu Kanai and Tae Kamakura
Department of Neonatology, Ibaraki Children's Hospital
Abstract
Objective: Lung ultrasonography was reported as a useful tool for diagnosing several lung diseases. However, there are no reports of lung ultrasonography for neonates. To date, neonatal pneumothorax was diagnosed by X-ray examination or transillumination. In this study, we evaluated the usefulness of ultrasonography for diagnosis of neonatal pneumothorax.
Methods: The study included 20 patients with proved pneumothorax and 20 patients in whom we excluded pneumothorax by chest radiography. Four neonatologists who did not perform the ultrasonography reviewed the records of ultrasonography.
Results: The "Lung Point" was observed and "Lung Sliding" was absent in all pneumothorax patients. However, some cases did not present the "Stratosphere Sign". In this study, the sensitivity in diagnosing pneumothorax was 100%, specificity 100%, positive predictive value 100%, and negative predictive value 100% via B-mode. Via M-mode, the sensitivity was 78%, specificity 81%, positive predictive value 83%, and negative predictive value 77%.
Conclusion: Lung ultrasonography is an accurate and reliable method to diagnose neonatal pneumothorax.




Original Article
Title
The Need to Investigate Urological Abnormalities in Acute Epididymitis in Children
Author
Chinatsu Onodera1) Nobuyuki Yotani1)2) Akira Nagai1) Mikiko Miyasaka3) and Akira Ishiguro1)2)
1)Department of General Pediatrics & Interdisciplinary Medicine, National Center for Child Health and Development
2)Center for Postgraduate Education and Training, National Center for Child Health and Development
3)Department of Radiology, National Center for Child Health and Development
Abstract
Acute epididymitis in young children often stems from congenital anomalies of the kidney and urinary tract (CAKUT). However, the need to investigate CAKUT in such patients is not widely recognized. We retrospectively examined males under age 18 years who received the diagnosis of acute epididymitis between April 2002 and November 2012 at the National Center for Child Health and Development, a tertiary pediatric hospital. The diagnoses were based on the symptoms, physical findings, and ultrasonography by radiologists. Of 105 patients between 3 months and 17 years of age with acute epididymitis, 17 patients, including four out of five with recurrent epididymitis, had underlying CAKUT. Sixteen patients with an urinary tract infection were younger and had a higher rate of CAKUT than 89 patients without an urinary tract infection. A voiding cystourethrography was performed for five infants under age one year with acute epididymitis. Among these patients, four had urethral stenosis, and three received an internal urethrotomy. Younger boys with acute epididymitis tended to have an urinary tract infection. Urological examinations for CAKUT, including a voiding cystourethrography, should be considered for the first episode of acute epididymitis in infants to evaluate urethral health.




Original Article
Title
Effectiveness of Rotavirus Vaccines for Decrease of Rotavirus Gastroenteritis in a Pediatric Outpatient Clinic, Analyzed by Vesikari Score
Author
Shuji Nakata1) Takeshi Tsugawa2) and Mayumi Ono2)
1)Nakata Pediatric Clinic
2)Department of Pediatrics, Sapporo Medical University
Abstract
To assess the efficacy of rotavirus (RV) vaccines in reducing office visits for RV gastroenteritis (RVGE), fluctuations in the number of RVGE cases were analyzed in one pediatric clinic in Sapporo during 7 years before and after vaccine introduction. We collected 1,574 stool specimens from patients aged mainly younger than 5 years old visiting the clinic between January 2005 and September 2018 and tested them for RV antigen by immunochromatography. The RV-positive samples were analyzed for G/P/I genotypes by RT-PCR. Clinical severity of the patients was evaluated by Vesikari score between the 2013 to 2018 seasons. Whereas the RV-positive number decreased after the vaccine uptake rates reached to 50% or more in the 2014 season, further reduction was not observed when the vaccine uptake rates reached about 70%. The rate of RVGE patients who received a full dose of RV vaccines increased from 15% to 65% between the 2014 and 2018 seasons. They were not hospitalized and the rate of intravenous drip infusion diminished among them. The severity of disease in the vaccinated cases was statistically mild compared with that in the unvaccinated cases by Vesikari score. Indirect protection to unvaccinated populations provided by RV vaccination was suggested and the severity of disease in the unvaccinated cases was reduced. The variety of RV G/P/I genotypes was detected from year to year. The efficacy of RV vaccine to reduce office visits for RVGE and to decrease the severity of disease was objectively demonstrated.




Case Report
Title
Three Cases of Percutaneous Closure for Congenital Portosystemic Shunt with Hypergalactosemia
Author
Shoji Takahashi Takashi Higaki Toshiyasu Ito Haruka Iwata Toyohisa Miyata Ryusuke Watanabe Tomozou Moritani Masaaki Ota Hidemi Takata and Eiichi Ishii
Department of Pediatrics, Graduate School of Medicine, Ehime University
Abstract
Congenital portosystemic shunt (CPSS) causes various complications because congenital abnormal blood vessels transport portal blood, without being metabolized in the liver, directly to the systemic circulation. CPSS is seen more frequently than hereditary enzyme deficiency in diseases causing hypergalactosemia detected by neonatal mass screening. It is most likely to be asymptomatic, and that makes diagnosis very difficult. CPSS may cause severe complications such as pulmonary hypertension, liver cirrhosis, liver tumor, liver pulmonary syndrome, hepatic encephalopathy, and accumulation of manganese in the basal ganglia. It is vital, therefore, to diagnose and treat it as soon as possible. Recently, percutaneous treatment using coils or vascular plugs has become common, and it is less invasive and more feasible than conventional methods: surgical operations to treat CPSS. We report 3 cases of hypergalactosemia, detected by mass screening and which we diagnosed as CPSS. We successfully treated all the cases by percutaneous closure before they showed symptoms and complications.




Case Report
Title
A sibling case of nephrogenic diabetes insipidus with different developmental progress depending on the time of treatment intervention
Author
Takuma Matsuki Naonori Kumagai Nao Uchida Hiroki Kudo Takaaki Kigoshi Masato Nihei Chisumi Sogi Sayaka Kawashima Dai Suzuki Miki Kamimura Natsuko Ichinoi Junko Kanno and Shigeo Kure
Department of Pediatrics, Tohoku University Hospital
Abstract
Congenital nephrogenic diabetes insipidus (CNDI) is a rare genetic disorder, and affected individuals present with not only polyuria and polydipsia, but also other conditions such as failure to thrive, intellectual disability, and intermittent fever, among others. Although there is currently no fundamental therapeutic strategy for CNDI, early intervention could help prevent failure to thrive, and even irreversible central neurological disorders. Therefore, asking about an individual's family history is very important. Here we present a report of two brothers with CNDI. The elder presented with failure to thrive and developmental coordination disorder, but he had not undergone a detailed examination in his first year of life. Several examinations in our hospital revealed his extremely low urinary osmotic pressure and extremely high serum arginine vasopressin, then he was diagnosed with CNDI. Thiazide diuretics and non-steroidal anti-inflammatory agents were effective against his polyuria, and therapeutic interventions improved his growth and motor development. The younger was born after the elder had received his diagnosis. The family had the younger admitted to our hospital because of poor weight gain. An examination revealed that he had the same genetic mutation as his brother, so thiazide diuretics and low-salt milk were started. The intervention improved his growth, similar to his elder brother. Developmental coordination disorder was not seen in the younger.




Case Report
Title
A Comparison of Cytokines in a Child with Acute Focal Bacterial Nephritis with Mild Encephalopathy and Acute Pyelonephritis
Author
Hiroyuki Tomino1)2) Masafumi Oka1)2) Yasufumi Ohtsuka1) Masafumi Zaitsu1)2) Hiroyuki Nunoi3) and Muneaki Matsuo1)
1)Department of Pediatrics, Faculty of Medicine, Saga University
2)Department of Pediatrics, Ureshino Medical Center
3)Department of Pediatrics, Reproductive and Developmental Medicine Course, Miyazaki University
Abstract
Acute focal bacterial nephritis (AFBN) is an inflammatory mass lesion due to local bacterial infection of the kidney parenchyma without obvious abscess formation. It is characterized by a lower frequency of pyuria and higher frequency of vesicoureteral reflux than acute pyelonephritis (APN). There have been several cases of AFBN with mild encephalitis / encephalopathy with a reversible splenial lesion (MERS), and a number of reports have investigated the pathophysiological mechanisms by measuring cytokines at the time of AFBN and/or MERS. We measured levels of several cytokines in the serum and cerebrospinal fluid of a patient suffering from AFBN with MERS, and APN. This one-year-old boy was hospitalized with AFBN accompanied by MERS. One and a half months after discharge, he suffered from APN after voiding cystourethrogram. The levels of IL-6, IL-10, IFN-γ and TNF-α in the serum during AFBN with MERS were lower than those with APN alone. Furthermore, the levels of these cytokines in the cerebrospinal fluid were higher than those in the serum. The elevation of cytokines in the cerebrospinal fluid compared with the serum might be related to the mechanisms underlying the onset of MERS from AFBN.




Case Report
Title
A Case of 46, XX Disorders of Sex Development Due to Maternal Adrenal Adenoma
Author
Tomoyuki Tani1) Keisuke Nagasaki1) Hiromi Nyuzuki1) Sunao Sasaki1) Yohei Ogawa1)2) and Akihiko Saitoh1)
1)Department of Pediatrics, Niigata University Graduate School of Medical and Dental Sciences
2)Department of Community Medicine, Niigata University Graduate School of Medicical and Dental Sciences
Abstract
The 46, XX disorders of sex development (DSD) can be classified into disorders of gonadal development, androgen excess during pregnancy, and disorders of internal/external genital development. Most cases of androgen excess are caused by fetal androgen excess such as a 21-hydroxylase deficiency, and only a few cases are due to maternal androgen excess. Here, we describe the case of a patient who was 3 months old at her first examination. She was born at 30 weeks of gestation via emergency cesarean section due to worsening maternal pregnancy-induced hypertension. The abnormal external genitalia (clitoral hypertrophy, labial fusion urogenital sinus, and non-palpable gonad) was recognized at birth. Her karyotype was SRY-negative 46, XX. Magnetic resonance imaging revealed a uterus in the pelvis and a vaginal structure, so her social sex was determined to be female. A neonatal screening test revealed no abnormalities. Fetal androgen excess was suspected because her mother had virilizing signs during pregnancy. However, this was judged to be unlikely based on her urine steroid profiles. We again focused on her mother and found that virilization had been persistent before pregnancy and after delivery. Moreover, the mother had an increased serum dehydroepiandrosterone sulfate level and a functional tumor in her right adrenal gland, which was later removed by laparoscopic surgery. The tumor histology indicated an adrenal adenoma. Overall, physicians must always keep in mind that androgen excess during pregnancy could cause 46, XX DSD, and therefore, should examine the mother carefully and conduct a detailed interview.




Case Report
Title
Acute Focal Bacterial Nephritis Caused by Staphylococcus aureus in a Boy with Atopic Dermatitis
Author
Yuta Asakura Katsuhiko Kitazawa Akihito Honda Masayoshi Senda Hironobu Kobayashi Akihiro Iguchi Mariko Arakawa and Kyosuke Ibi
Department of Pediatrics, Asahi General Hospital
Abstract
Staphylococcus aureus (S. aureus) rarely causes febrile urinary tract infection (FUTI) in children. We report a case of acute focal bacterial nephritis (AFBN) caused by methicillin-susceptible Staphylococcus aureus (MSSA).
An 8-year-old boy with atopic dermatitis (AD) was admitted to our hospital because of fever for 7 days. He had no medical history of FUTI. His skin was dry, with widespread excoriated erythema in the extremities. A midstream urine specimen revealed pyuria; the Gram-stained smear showed Gram-positive cocci. A presumptive diagnosis of FUTI was made, and antimicrobial therapy with cefotaxime and ampicillin was initiated. On hospital day 3, he remained febrile. Contrast-enhanced computed tomography of the abdomen revealed multiple tumor-like low-density lesions in the left kidney, confirming the diagnosis of AFBN. Urine culture was positive for MSSA. Based on the results of antimicrobial susceptibility testing, treatment was changed to intravenous cefazolin (CEZ), and his fever abated within two days. CEZ was given for a total of 3 weeks. Voiding cystourethrography showed bilateral vesicoureteral reflux (VUR).
In this patient, it seems likely that MSSA colonizing the skin caused AFBN, although its pathogenesis was not determined. S. aureus should be considered as a possible pathogen in FUTI, including AFBN, in children with urinary tract abnormalities such as VUR, especially when accompanied by active AD.




Case Report
Title
Three Cases of Drug Intoxication False-negative on a Handy Drug Screening Kit
Author
Shogo Otake1) Kosuke Kohashi1)2) Hiroshi Okada1) and Ryugo Hiramoto1)
1)Department of Pediatrics, Matsudo City General Hospital
2)Education and Research Center of Legal Medicine, Graduate School of Medicine, Chiba University
Abstract
A handy drug screening kit, Triage DOA (SYSMEX CORPORATION, Japan), can be used to easily detect various drugs without any special equipment, and is often used in many emergency departments. There have been several reports of false-negative results for certain drugs because of drug detection insensitivity, while no pediatric case series has been reported. We encountered three cases of drug intoxication false-negative for Triage DOA. Although Triage DOA gave negative results on admission in all cases, we strongly suspected drug intoxication from the clinical history and family background. We performed drug screening by mass spectrometry and causative drugs, flunitrazepam and etizolam, were detected. These three cases indicated two important points. First, Triage DOA can give false-negative results in pediatric cases, and second, the results tended to be false-negative for flunitrazepam and etizolam in particular. In conclusion, we should consider drug intoxication detectable by Triage DOA based on a detailed clinical history and family background and perform drug screening by mass spectrometry, even if Triage DOA gives negative results in patients with disturbance of consciousness.




Case Report
Title
A Case of a Transfer from a Secondary or Tertiary Medical Institution to Home Care through a Home Care Support Hospital
Author
Mizuki Shibuya1) Yoshimi Nishizaki1) Mari Hagio1) Yoshiko Iwamaru1) Kumiko Inoue1) Daisuke Sato1) Maiko Igarashi1) Seichi Matsumura1) Tomomi Naito1) Hiroko Shima1) Takuma Ishii1) Shoichi Oyama1) and Akihito Tsujikawa2)
1)Department of Pediatiatrics, Saiseikai Kawaguchi General Hospital
2)Tsujikawa Home Clinic
Abstract
Problems and strategies for improvement in home care transfer for children dependent on medical care were examined through analysis of a case at this hospital. It was found that secondary/tertiary medical institutions and home care support hospitals have different opinions regarding home care transfers. Beneficial cooperation was found to be possible through establishing several opportunities for information sharing between these two facilities and the home care support clinic. It was also found to be important to actively request participation in conferences from the municipality and local fire station. Further, each facility was able to operate efficiently with all information in one location because the home care support department staff at the home care support hospital took on a leading role in the casework. Thus, the home care support department staff were considered suitable substitutes for care managers in adult treatment.




Brief Report
Title
Current Risk Assessment of Nicotine Poisoning from Ingestion of Heat-not-burn Tobacco: Eight Pediatric Cases
Author
Yuta Sasaoka Hirokazu Takei Osamu Nomura and Yusuke Hagiwara
Division of Pediatric Emergency Medicine, Tokyo Metropolitan Children's Medical Center
Abstract
<Background>
Pediatric cases of ingestion of heat-not-burn (HnB) tobacco products were first reported in Japan in 2016, but their clinical features are still unknown. The purpose of this study was to clarify the clinical features of pediatric cases of HnB tobacco product ingestion.
<Methods>
This was a retrospective case series enrolling 8 patients with HnB tobacco ingestion who visited the pediatric emergency department at Tokyo Metropolitan Children's Medical Center between April 2016 and August 2017.
<Results>
The mean age of the patients was 11.5 months. Only 1 patient experienced vomiting due to tobacco ingestion. Four patients had ingested unused sticks and the remaining four patients had ingested HnB cigarette butts.
<Discussion>
Inadvertent ingestion of HnB tobacco products can lead to acute nicotine poisoning. However, the current data are insufficient to estimate the amount of tobacco ingestion that would cause acute nicotine poisoning in infants or the risk of ingesting HnB cigarette butts.




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