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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.123, No.5, May 2019
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Original Article
Title
Effectiveness of Topical Steroids on Phimosis in Early Infancy
Author
Masahiro Hiraoka
Aiiku Pediatric Clinic
Abstract
The incidence of urinary infection is high in early infancy, especially in boys with phimosis. Effectiveness of topical steroids on phimosis were examined in early infancy.
The subjects consisted of 187 infants who were brought to our clinic for routine vaccination at the age of two months. Phimosis was graded as 0 to 4 according to the retractility of the prepuce with a gentle retraction maneuver. Phimosis 1 to 4 was further classified by another retraction maneuver with moderate strength as phimosis A when completely retracted, and as phimosis B when not completely retracted. Phimosis A was treated by hydrocortisone ointment twice daily, and infants with phimosis B were treated by betamethasone ointment, until the prepuce showed no redness after retraction.
Phimosis B was identified in 19 infants, being significantly more frequent in infants with phimosis 4 (15 of 84, 17.9%) than in those with phimosis 1 to 3 (4 of 95, 4.2%). More infants with phimosis B required topical steroid treatment for four weeks or more than those with phimosis A. The prepuce was fully retracted in 79.9% of the infants examined at the age of three months, and 91.6% of those at the age of one year. The prepuce was fully retracted in more infants with phimosis 1 to 3 at the age of one year than those with phimosis 4 (97.6% vs. 84.7%, respectively).
Topical steroid therapy was easy and effective for phimosis in early infancy, while severe phimosis required longer treatment than mild phimosis.
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Original Article
Title
Fall Injuries in Infants while the Parent is Operating a Bicycle
Author
Osamu Nomura1)3) Yusuke Miyazaki2) Hirokazu Takei1) Mariko Terauchi1) Shun Kishibe1) Yusuke Hagiwara1) Koji Kitamura3) Yoshifumi Nishida3) and Tatsuhiro Yamanaka3)4)
1)Division of Pediatrics Emergency Medicine, Tokyo Metropolitan Children's Medical Center
2)Tokyo Institute of Technology, Dept. of Systems and Control Engineering
3)National Institute of Advanced Industrial Science and Technology (AIST) Artificial Intelligence Research Center
4)Ryokuen Children's Clinic
Abstract
Background
Pediatric emergency physicians commonly experience cases of trauma in infants resulting from the child falling from the arms or the back of a parent while the latter is operating a bicycle.
Methods
1. We conducted a retrospective case-series study enrolling children younger than 1 year of age who were injured by falling from the arms or the back of the parent while the latter was operating a bicycle.
2. We conducted a dynamics-experiment by recreating the circumstances of the accident using dummies representing a 6-month-old infant being carried on the back of the mother. We assessed the Head Injury Criterion (HIC) score and the maximum impact load on the head of the dummy infant.
Findings
1. We found 8 injured patients, 2 of whom required intensive care. One of them experienced neurological sequelae.
2. The HIC scores and the maximum impact load varied 7.7 to 17.0 and 2.26 to 3.47 times the reference values for 6-month-old infants, respectively.
Conclusions
Our study revealed that the strong impact on the infants' head due to this mechanism could result in severe head injuries. Preventive strategies for the safe transport of infants are needed.
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Case Report
Title
A Female Case of Pyruvate Dehydrogenase Complex Deficiency Presenting with Antenatal Brain Dysgenesis
Author
Emi Yanai1) Sakae Kumasaka1) Yusuke Terada1) Yosuke Akahane1) Osamu Kizumi1) Yoshio Shima2) and Etsuo Naito3)
1)Department of Neonatology, Japanese Red Cross Katsushika Maternity Hospital
2)Department of Neonatology, Nippon Medical School Musashikosugi Hospital
3)Department of Pediatrics Japanese Red Cross Tokushima Hinomine Rehabilitation Center for People with Disabilities
Abstract
A female baby was born weighing 1,828 g at 37 weeks' gestation by cesarean section. Agenesis of the corpus callosum and ventriculomegaly of the fetus were identified at 24 weeks' gestation. Dysmorphic facial features including low-set ears, cleft palate, mandibular micrognathia, finger overlapping, and rocker bottom feet were noted. The gradual development of apnea and lactic acidosis was observed with the initiation of breast feeding. Lactate and pyruvate levels in serum and cerebrospinal fluid were elevated (serum lactate/pyruvate 10.3/0.84 mmol/L, cerebrospinal fluid lactate/pyruvate 16.8/2.03 mmol/L). Mutational analyses were performed and a change in c.968_989dup (p.S331DfsX16) in the PDHA1 gene was detected. Lactic acidosis was improved by a ketogenic diet that was started in order to provide alternate energy. However, the patient developed recurrent apnea and EEG recordings showed hypsarrhythmia on day 112.
Although PDHC deficiency with anatomic brain abnormalities is frequently associated with West syndrome and severe lactic acidosis from the early neonatal period, particularly in female patients with the E1α mutation, antenatal brain dysgenesis is rarely reported. We present a female patient with E1α deficiency who demonstrated antenatal brain abnormality.
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Case Report
Title
Two Adolescent Cases of Muscular Dystrophy with Acute Myocardial Infarction
Author
Aiko Kumamoto Takashi Kumamoto Katsuya Tashiro and Muneaki Matsuo
Department of Pediatrics, Faculty of Medicine, Saga University
Abstract
In muscular dystrophy (MD), it is assumed that myocardial damage preferentially occurs in the inferolateral wall of the left ventricle, and mostly proceeds to dilated cardiomyopathy.
Herein we report on two adolescent cases of MD in which coronary spasm appeared to be involved in myocardial infarction (MI) of the inferolateral wall.
Case 1 was a 12-year-old boy with Becker-type MD. At 8 years of age, the patient started experiencing chest pain. An electrocardiogram (ECG) revealed ST segment elevation in Leads II, III, aVF, and V5-6; in addition, the lower wall of the left ventricle (LV) was hypokinetic. There was no stenosis on coronary angiography (CAG). At 12 years of age, the patient experienced similar attacks of chest pain, and similar changes were seen on an ECG. CAG revealed narrowing throughout the left coronary artery and marked narrowing of the circumflex branch.
Case 2 was a 14-year-old boy with Duchenne-type MD. The patient experienced chest pain once. An ECG revealed ST segment elevation in Leads II, III, aVF, and V5-6; the posterolateral wall of the LV was hypokinetic. There was no stenosis on CAG. Spasticity of the right coronary artery was seen during the recovery period of the acetylcholine loading test.
When a patient with MD suffers myocardial injury, increased vascular endothelial function should also be considered, with the early introduction of Ca antagonists which can possibly be effective in suppressing myocardial disorders.
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Case Report
Title
A Case Report of Recessive Hereditary Methemoglobinemia Type I in Siblings
Author
Youichi Haga1) Hiroyuki Takahashi1) Kazumasa Mitsui1) Toshinori Nakamura2) Masaki Matsuoka1) Yasuko Kojima1) Hiromi Ogura3) Hitoshi Kanno3) Yukio Hattori4) and Akira Ohara1)
1)Department of Pediatrics, Toho University Omori Medical Center
2)Department of Pediatrics, Showa University School of Medicine
3)Department of Transfusion Medicine and Cell Processing, Tokyo Women's Medical University
4)Department of Clinical Laboratory, Yamaguchi Saiseikai General Hospital
Abstract
Hereditary methemoglobinemia is divided into two categories, namely, methemoglobinemia due to abnormal hemoglobin variants such as hemoglobin M, and from NADH-cytochrome b5 reductase enzyme deficiency (cyt b5R deficiency). cyt b5R deficiency causes the accumulation of methemoglobin due to decreased NADH reductase activity, which then disturbs the conversion of methemoglobin to hemoglobin. Recessive hereditary methemoglobinemia has been classified into four subtypes. This report presents siblings with hereditary methemoglobinemia type I. Sibling A: a 7-year old boy was given a diagnosis of methemoglobinemia (Met Hb 14.0%) after developing pneumonia and febrile seizures. Sibling B: a 5-year old girl was given a diagnosis of (Met Hb 12.0%), when she also developed pneumonia. Both siblings had diarrhea and poor weight gain for several months after birth. The cyt b5R activity levels in their erythrocytes were markedly low. We discovered a point mutation (p.Arg57Gln) in the cyt b5R gene in both cases, indicative of type I recessive hereditary methemoglobinemia. The patients were treated with oral ascorbic acid and riboflavin. Their current methemoglobin levels have since stabilized at approximately 7%.
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Case Report
Title
Clinical Value of Urinary Mulberry Bodies in Diagnosing Fabry Disease
Author
Takafumi Fukuda Yasuhiro Ikawa Takuya Mimura Ryo Nakagawa Maiko Takakura Shimpei Yamada Tetsujiro Shirahashi Masaki Shimizu Michiko Okajima and Akihiro Yachie
Department of Pediatrics, Kanazawa University Hospital
Abstract
Fabry disease (FD) is a lysosomal disorder in which irreversible fatal organ dysfunction develops after adolescence. Therefore, the early initiation of enzyme replacement therapy is indispensable to improve patient prognosis. Pediatricians play an important role in early diagnosis because neuropathic pain in the extremities is manifested during childhood. However, concluding a diagnosis based on the symptoms remains problematic. Here, we describe three FD patients who were diagnosed by the detection of urinary mulberry bodies (UMB).
Case 1. A 13 year-old-boy who had endured lower extremity pain for three years presented to the hospital. Detection of UMB led to the suspicion of FD. This diagnosis was confirmed by the finding of low alpha-galactosidase A activity.
Case 2. The mother of the above patient had proteinuria and hypertrophic cardiomyopathy of unknown etiology for several years. The diagnosis of her son prompted her to undergo tests, which revealed that she also had UMB, indicating a diagnosis of FD.
Case 3. A 13-year-old girl presented with cornea verticillata. This characteristic finding suggested the possibility of FD, which was confirmed by the detection of UMB.
Despite having different chief complaints, all 3 patients had UMB and were given diagnoses of FD. Since urinary sedimentation can be determined at any hospital, urinalysis should be performed when FD is suspected.
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Case Report
Title
A Case of Negative Pressure Pulmonary Edema Resulting from Misuse of a Child Safety Seat
Author
Kentaro Kogawa1)2) Ryo Matsuoka1)2) Kenji Yoshida1)2) Tomoko Hara1) Satoshi Tonezawa1) Ryosuke Nambu1) Shin-ichiro Hagiwara1) Seiichi Kagimoto1)2) and Hiroyuki Ida2)
1)Division of General Pediatrics, Saitama Children's Medical Center
2)Department of Pediatrics, The Jikei University School of Medicine
Abstract
Negative pressure pulmonary edema (NPPE) is a noncardiogenic pulmonary edema that results from the generation of high negative intrathoracic pressure to overcome upper airway obstruction (UAO). Young adults have an increased risk of NPPE in the postoperative setting. This is the first reported case of NPPE caused by misuse of a child safety seat.
A previous healthy 1-month old infant had sudden cyanosis while he was sitting in a child safety seat. Chest radiography revealed bilateral changes consistent with pulmonary edema, but transthoracic echocardiography demonstrated normal function. His respiratory status improved with mechanical ventilation, and he was discharged on hospital day 9. We diagnosed NPPE because of UAO, rapid onset, pink frothy sputum and a rapidly resolving clinical course. We speculate that NPPE resulted from misuse of the child safety seat. After we guided his parents on the correct use of the child safety seat, no recurrence was reported.
NPPE rarely develops in daily life, and it is difficult for pediatricians to diagnose. Symptoms improve promptly with appropriate respiratory care; therefore, it is a matter of concern that we pay no attention to diagnosis it and search for the cause. Our awareness of NPPE will contribute to the prevention of recurrence. Education in the proper way of using a child safety seat is also important because NPPE is likely to result from its misuse.
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Case Report
Title
Two Cases of Children with Diphenhydramine Intoxication Due to Massive Oral Ingestion of a Marketed Drug
Author
Masanori Ishimaru1) Kota Hirai1)2) Satoshi Takasago2)3) Mariko Ikegami2) Hideyuki Tabata1) Shinichi Matsuda1) Masahiko Kato1) and Hiroyuki Mochizuki1)
1)Department of Pediatrics, Tokai University School of Medicine
2)Department of Pediatrics, Tokai University Hachioji Hospital
3)Division of Cardiology, Tokyo Metropolitan Children's Medical Center
Abstract
Diphenhydramine (DPH) is a first-generation H1-antagonist and is easily obtained as a commercial agent. Case 1 was a 13-years-old girl who presented with consciousness disorder as a chief complaint and was initially treated under suspicion of encephalitis or encephalopathy. Later, a large amount of Travelmin® was found at her home. QT prolongation was detected which may be induced by DPH intoxication. Case 2 was a 14-years-old girl with consciousness disturbance and convulsions. Initially, encephalitis/encephalopathy was suspected. Poisoning was later suspected after the appearance of arrhythmia, such as QT prolongation and VF. Many empty boxes of Drewell® were found at her home, and drug intoxication was diagnosed based on high blood concentrations of DPH. These over-the-counter medicines are often used as available medications for suicide. The number of cases of intoxication with these agents may increase in the future, even among children, given the ease of browsing the Internet for information. As DPH cannot be detected by a simple urinary drug test, caution should be employed in cases of such intoxication.
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Brief Report
Title
Survey Result of Antibiotic Prescribing Behavior toward Children in Himeji-city Emergency Medical Center for Nights and Holidays
Author
Shota Myojin1) Naohiro Kamiyoshi1) Masaaki Kugo1) Akihiro Hongo2) and Masashi Kasai3)
1)Japanese Red Cross Society Himeji Hospital, Department of Pediatrics
2)Hongo Children's Clinic
3)Hyogo Prefectural Kobe Children's Hospital, Department of Pediatric Infectious Diseases
Abstract
The Japanese government adopted a national action plan on antimicrobial resistance in 2016; reducing antimicrobial use in 2020 to two-thirds of the level in 2013. Himeji-city Emergency Medical Center for Nights and Holidays (HEMCNH) is the hub medical institution run mainly by private physicians. Therefore, the information about antibiotics prescribing behavior in this center provides useful hints to promote antimicrobial stewardship at the community level. We aimed to analyze antibiotic prescribing behavior toward children in HEMCNH. Primary care data of children <15 years of age recorded from September 2014 to March 2018 in the Himeji Emergency Medical Service Association database was analyzed. Records about patients' age, diagnoses, and antibiotics prescribed were extracted. We used days of therapy/1,000 patients-visits (DOTs) for evaluation. Antibiotics were prescribed for 13% of patients, and 17% with acute respiratory tract infections. Oral 3rd-generation cephalosporins were frequently prescribed regardless of age and clinical condition. This study has identified substantial overprescribing of antibiotics in pediatric primary care, particularly in respiratory tract conditions. There is a large potential to lower antibiotic prescribing in this area.
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