gakkaizashi


THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.125, No.6, June 2021


Review
1. The Timing of Discontinuation of Non-invasive Repositioning from the Viewpoint of Intussusception Treatment in Our Hospital
Original Article
1. Assessment of Relationship Between Iron-deficiency Anemia and Febrile Seizures in Infants with Exanthema Subitum
2. Clinical Course and Final Diagnosis of Thrombocytopenia in Childhood
3. The Loop-mediated Isothermal Amplification Assay for Definitive Diagnosis of Mumps in Vaccinated Children
Case Report
1. IgA-vasculitis Complicated by Ureteritis and Urolithiasis
2. A Case of Cystic Fibrosis Diagnosed by Detection and Scrutiny of Cystic Fibrosis-associated Liver Disease
3. Refractory Kawasaki Disease Treated with Centrifugal Therapeutic Plasma Exchange: A Comparison with Membrane Filtration Method
4. A 12-year-old Boy, Belonging to a Sumo Club, with Type 2 Diabetes Mellitus Requiring Insulin Replacement Therapy
5. An Early Infant with Pertussis Showing Symptom Recurrence and Bacterial Re-isolation after Clarithromycin Treatment
6. Pneumonia with Bacteremia Caused by Cronobacter sakazakii in an Infant
7. Three Cases of Subgaleal Hematoma Due to Hair Pulling


Review
Title
The Timing of Discontinuation of Non-invasive Repositioning from the Viewpoint of Intussusception Treatment in Our Hospital
Author
Yuki Sakaguchi Satoko Tsuchida and Masamichi Tamura
Department of Pediatrics, Japanese Red Cross Akita Hospital
Abstract
Intussusception is one of the most typical causes of pediatric acute abdomen. We reviewed cases of intussusception in our institution from the last decade (August 2008 to September 2018) and compared their management, focusing on reduction time, with that of cases reported in the literature and guidelines for intussusception management published by the Japanese Society of Emergency Pediatrics.
We identified a total of 22 cases (14 males [64%] and 8 females [36%]). Regarding the age of onset, 18 patients (81.8%) were <2 years old. Radiologic hydrostatic reduction was performed for all patients except in 2 cases, which were initially indicative of surgical reduction. The hydrostatic reduction was successful in 15 cases (group A, 75%), while it was discontinued in the remaining 5 cases (group B, 25%). There were no significant differences between the two groups in age, the interval from onset to the first visit, pressure used for hydrostatic reduction (initial and maximum), and rate of sedative usage. Conversely, the procedure time was significantly longer in group B (6-68 minutes; median, 53 minutes) than in group A (2-52.5 minutes; median, 11.2 minutes). Furthermore, 14 (93.3%) patients in group A underwent a successful reduction in <30 minutes, while 4 (80%) patients in group B demonstrated procedure time of ≥30 minutes but without good results.
In conclusion, our findings suggest that successful hydrostatic reduction time was generally within 30 minutes. If this period is exceeded, it is necessary to consult a pediatric surgeon as soon as possible, while considering the possibility of surgical reduction.




Original Article
Title
Assessment of Relationship Between Iron-deficiency Anemia and Febrile Seizures in Infants with Exanthema Subitum
Author
Yoshinari Inoue1) Kazushi Tamura2) and Takeshi Tomomasa3)
1)Inoue Pediatric Clinic
2)Tamura Pediatric Clinic
3)Pal Pediatric Clinic
Abstract
The association of iron-deficiency anemia (IDA) with the pathology of febrile seizure (FS) has been reported. We consecutively enrolled 407 infants who suffered from exanthema subitum (ES) that is susceptible to FS. They were divided into two groups by the presence of FS complication: 33 FS (+) and 374 FS (−). Complete blood counts were tested in capillary blood during the febrile stage. The demographic comparison of the two groups revealed that the FS (+) group had a higher frequency of FS family history than FS (−) group [54.5 vs. 13.1%, P=0.000], and a higher age in months by median (interquartile range) [16 (13—20) vs. 13 (10—17) months, P=0.003]. However, no significant difference was noted in hemoglobin (Hb) or mean corpuscular volume (MCV). By use of Hb<11 g/dL, hematocrit<33 % and MCV<70 fL as criteria for IDA, incidence of IDA was 0 in FS (+) vs. 26 in FS (−). Multiple logistic regression analysis was performed by setting FS complication as an object variable and the family history, age in months, and MCV as explanatory variables. Only the family history was highly significant (P ≈ 0; odds ratio 11.2 with 95% CI of 5.2—24.9), while MCV showed no association. These findings suggest that, although FS complication in ES showed strong association with the family history of FS, its association with the presence of IDA is unlikely.




Original Article
Title
Clinical Course and Final Diagnosis of Thrombocytopenia in Childhood
Author
Yuji Miyajima Tomoki Ueda Nao Hatashita Chika Yasufuku Konomi Suzuki Sho Narahara Daisuke Omori Tatsuya Fukasawa and Tetsuo Kubota
Department of Pediatrics, Anjo Kosei Hospital
Abstract
We explored the clinical courses and final diagnoses of 172 children with thrombocytopenia. A total of 158 children (91.9%) developed idiopathic thrombocytopenic purpura (ITP), 10 (5.8%) developed hereditary thrombocytopenia, and 4 (2.3%) developed other diseases (2 myelodysplastic syndrome and 2 collagen diseases) after initial diagnosis of ITP. The percentage of non-ITP patients increased to 34.1% among 41 patients who had thrombocytopenia for over 1 year, and to 42.4% among 33 patients who exhibited persistent thrombocytopenia. When the proportions of non-ITP patients were compared in terms of the platelet count at diagnosis, 1 of 140 patients (0.8%) was a non-ITP patient among those with platelet counts less than 2.0×104/μL, whereas 13 of 49 patients (26.6%) were non-ITP patients among those with platelet counts more than 2.0×104/μL. When the proportions of non-ITP patients were compared by age at diagnosis, 8 of 156 patients (5.1%) were non-ITP patients among those aged 9 years or younger, but 6 of 16 patients (37.5%) were non-ITP patients among those aged 10 years or older. As 8.1% of children with thrombocytopenia were diagnosed with conditions other than ITP, differential diagnosis of hereditary thrombocytopenia requires confirmation of a relevant family history and examination of platelet morphology. Furthermore, it is necessary to consider whether the diagnosis should be changed even after diagnosis of ITP.




Original Article
Title
The Loop-mediated Isothermal Amplification Assay for Definitive Diagnosis of Mumps in Vaccinated Children
Author
Kensei Gotoh Naoko Nishimura Arika Murase Mako Yamada Takuma Ando Takuya Akano Takuto Ito Suguru Takeuchi Kyogo Suzuki Koji Takemoto and Takao Ozaki
Department of Pediatrics, Konan Kosei Hospital
Abstract
Laboratory diagnostic methods are necessary for a definitive diagnosis of mumps in vaccinated children, but have not been adequately studied in Japan. In this retrospective study, we analyzed the clinical features of vaccinated children with laboratory-confirmed mumps. Subjects were 110 children who, presented with salivary gland swelling at our hospital between September 2013 and April 2017. We obtained salivary swabs for RNA detection using loop-mediated isothermal amplification (RT-LAMP) assay and serum samples for the detection of mumps IgG and IgM enzyme immunoassay antibodies. Laboratory confirmation of mumps was defined by either the detection of mumps virus RNA or IgM antibody of mumps virus. Of the 110 children, 59 children (median age, 6.0 years; range, 2.1—14.1 years) with a record of a single mumps vaccination (median interval from vaccination, 3.1 years; range, 0.2—9.3 years) were analyzed.
Of the patients with clinically diagnosed mumps, 19 (32%) had laboratory-confirmed mumps. Among them, 19 (100%) were RT-LAMP positive and 3 (16%) were IgM positive. All 19 children were IgG positive, which was conjectured to be due to secondary vaccine failure. In a multivariate analysis, the proportions of patients around an epidemic (odds ratio, 9.4; 95% CI, 1.7—53.8), and patients with acute inflammation of the submaxillary gland (odds ratio, 5.1; 95% CI, 1.1—24.5) were significantly higher in the laboratory-confirmed mumps group than in the non-mumps group.
We conclude that the RT-LAMP assay is useful for a definitive diagnosis of mumps in vaccinated children presenting with salivary gland swelling.




Case Report
Title
IgA-vasculitis Complicated by Ureteritis and Urolithiasis
Author
Kohei Tonosono Katsuhiko Kitazawa Akihito Honda Masayoshi Senda Hironobu Kobayashi Akihiro Iguchi Chika Uchiyama Mariko Arakawa Yasuyuki Yamamoto Minjae Ma and Nahoko Satoh
Department of Pediatrics, Asahi General Hospital
Abstract
IgA-vasculitis is rarely associated with urological manifestations except nephritis. We report a case of a 7-year-old girl with IgA-vasculitis complicated by unilateral ureteritis and urolithiasis. She was hospitalized because of abdominal pain, petechiae on the lower extremities, and right knee joint pain, and was diagnosed with IgA-vasculitis on day 1. Right costovertebral angle tenderness and non-glomerular microhematuria were also notable. Abdominal ultrasonography (AUS) showed a duplicated renal pelvis, mild pyelectasis, and thickening of the ureteropelvic mucosal layers in her right kidney. Administration of prednisolone effectively relieved her skin and joint symptoms within one week but abdominal pain persisted intermittently. On day 12, after excluding a diagnosis of pyelonephritis, we diagnosed ureteritis complicating IgA-vasculitis based on AUS findings including further dilatation in the right pelvis. On day 20, she developed right flank pain and AUS revealed a stone in the right upper ureter. She was transferred to a specialized facility and underwent a transurethral lithotripsy 5 weeks later. In this case, we speculate that ureteritis associated with IgA-vasculitis, together with underlying duplicated pelvis and glucocorticoid (s)-induced hypercalciuria, might have contributed to stone formation. Attention should be given to the development of urolithiasis in children with IgA-vasculitis complicated by ureteritis, especially when treated with glucocorticoid (s).




Case Report
Title
A Case of Cystic Fibrosis Diagnosed by Detection and Scrutiny of Cystic Fibrosis-associated Liver Disease
Author
Shoichiro Kusunoki1) Rieko Sakamoto1) Suguru Watanabe1) Shogo Ito2) Takao Togawa2) and Kimitoshi Nakamura1)
1)Department of Pediatrics, Kumamoto University of Medicine
2)Department of Pediatrics, Nagoya City University Graduate School of Medical Science
Abstract
Cystic fibrosis (CF) is extremely rare in Japan and is not sufficiently recognized. It may be diagnosed as refractory bronchial asthma due to observed chronic respiratory distress. There are few reports of CF-associated liver lesions (CFLD) in Japan. Here, we report a case of CF diagnosed by accidental detection of CFLD. The patient was a 7-year-old boy. He was diagnosed with bronchial asthma from early childhood and had repeated acute exacerbations. At the time of acute exacerbation, a potato-like liver was found on chest CT, and he was referred to our hospital. In addition to liver lesions, sinusitis, allergic bronchopulmonary aspergillosis, and cystic pancreatic lesions were also reported. Histopathological examination of the liver revealed cholestatic cirrhosis due to bile duct mucus plugs. CF was strongly suspected, and a genetic test confirmed the diagnosis.
CF may be latent in children diagnosed with refractory bronchial asthma, and the detection of CFLD may be diagnostic. Imaging and pathological tests are more useful for detecting CFLD than clinical symptoms and biochemical tests. In refractory bronchial asthma with growth disorders and allergic bronchopulmonary aspergillosis, it is important to differentiate CF, scrutinize CFLD, and also consider sweat tests or genetic tests.




Case Report
Title
Refractory Kawasaki Disease Treated with Centrifugal Therapeutic Plasma Exchange: A Comparison with Membrane Filtration Method
Author
Norio Omori1) Masatomo Kitamura1) Shunsuke Noda1) Tsuyoshi Okamoto1) Norikazu Inamura1) Hironori Minoura1) Takahiro Saima1) Norimasa Kurosaka1) Toru Chigono2) Naoki Sato2) Shota Nakagawa2) Natsuki Minemura2) Haruka Obinata3) and Yohei Akazawa3)
1)Department of Pediatric Intensive Care, Nagano Children's Hospital
2)Department of Clinical Engineering, Nagano Children's Hospital
3)Department of Pediatric Cardiology, Nagano Children's Hospital
Abstract
Therapeutic plasma exchange (TPE) with membrane filtration is the mainstream TPE procedure in Japan, but centrifugal TPE is common in Europe and the USA. Here, we report a case of refractory Kawasaki disease treated with TPE using both membrane and centrifugal TPE. Specifically, a 3-month-old boy with refractory Kawasaki disease, including coronary artery dilatation, was started on TPE at day 9. Five treatments of TPE were conducted with 3 being membrane filtration and 2 involving the centrifugal method. In comparison, the IgG and fibrinogen reduction rate, which indicate the efficiency of TPE, were similar in both methods. However, the risk of clotting events was lower with membrane filtration TPE, whereas the treatment time was clearly shorter with centrifugal TPE. Several problems were encountered with membrane filtration TPE, such as blood removal failure and blockage of the separation membrane due to thrombus formation; in contrast, centrifugal TPE was performed smoothly without any difficulties. In conclusion, centrifugal TPE is an effective treatment method for infants with a low risk of clotting events, improved plasma removal efficiency, and shorter procedure time. Indeed, for infants who require appropriate vascular access and sedation, centrifugal TPE might be more useful than TPE with membrane filtration.




Case Report
Title
A 12-year-old Boy, Belonging to a Sumo Club, with Type 2 Diabetes Mellitus Requiring Insulin Replacement Therapy
Author
Akiko Yamamoto and Nagisa Komatsu
Department of Pediatrics, Kumamoto Chuo Hospital
Abstract
Heavyweight athletes, such as sumo wrestlers, make efforts to gain weight to improve their competitiveness. As harmful effects, visceral fat accumulation and hyperinsulinemia have been reported. Fatty liver and hyperlipidemia were observed even in growth-period heavyweight athletes. In this study, we report a 12-year-old boy, belonging to a sumo club, with type 2 diabetes mellitus.
The boy had belonged to a sumo club, achieving favorable results. He tended to be obese since 3 years of age. A health checkup indicated obesity when he was a fourth-year elementary school student, but he did not undergo detailed examination. Thirst, polydipsia, polyuria, and malaise were noted, and there was a 10 kg decrease in body weight in 2 weeks. He was taken to a hospital. The urinary glucose, ketone body, fasting blood glucose, and HbA1c values were 3+, 2+, 333 mg/dL, and 15.2%, respectively, suggesting diabetes mellitus. The boy was then referred to our hospital. Upon initial consultation, the height, body weight, and body mass index (BMI) were 170 cm, 92.3 kg, and 31.9 (99 percentile), respectively. Acanthosis nigricans was noted. The blood gas level was within normal range, and he was negative for autoantibodies. Based on his family history, a diagnosis of type 2 diabetes mellitus was made. After admission, insulin therapy was started. Self-secretion gradually returned, and the dose of insulin was decreased. Oral administration of metformin was started, and the boy was discharged. During admission, nutritional guidance was conducted for the boy and his mother, and has been continued at the outpatient clinic.
With respect to health-related problems in sports athletes, slimming related to sporting events, such as athletics and rhythmic gymnastics, has been emphasized, whereas few studies have reported health-related problems in heavyweight athletes, such as sumo wrestlers, especially in children. It may be necessary to evaluate the current status and promote regular, continuous health management.




Case Report
Title
An Early Infant with Pertussis Showing Symptom Recurrence and Bacterial Re-isolation after Clarithromycin Treatment
Author
Takuto Ito Naoko Nishimura Mako Yamada Takuma Ando Takuya Akano Kensei Gotoh Koji Takemoto and Takao Ozaki
Department of Pediatrics, Konan Kosei Hospital
Abstract
We present a case of a 31-day-old girl who was admitted with a three-day history of paroxysmal cough with cyanosis and apneic attacks. Pertussis was diagnosed by DNA detection and isolation of Bordetella pertussis. Clarithromycin (CAM) 15 mg/kg/day was administered for eight days, and the patient was discharged once symptoms had resolved. However, 7 days after the end of treatment, she required readmission for recurrence of paroxysmal cough and apneic attacks. Repeat testing showed DNA detection and isolation of Bordetella pertussis. Erythromycin (EM) at a dose of 30 mg/kg/day was administered for 14 days. Improvement of symptoms and negative DNA detection and isolation were confirmed following the EM course. Antimicrobial susceptibility testing with E-test was performed on two strains isolated at the time of initial admission and readmission, and they were susceptible to both CAM and EM. The source of infection was considered to be the father, who had previously received four doses of DPT vaccine, and CAM was administered on day 1 of initial hospitalization. During the initial hospitalization, the PT-IgG titer remained below 10 EU/mL, but at the time of readmission, increased to 27 mg/mL. Bordetella pertussis isolated during both the initial hospitalization and readmission was considered to be the same strain by genetic analysis. For pertussis in early infants, the 7-day course of CAM administration, as recommended in guidelines, may be inadequate.




Case Report
Title
Pneumonia with Bacteremia Caused by Cronobacter sakazakii in an Infant
Author
Hiroko Yokoyama1) Yuichi Kodama1) Yoshihiro Takahashi1) Satoshi Okada1) Tomohiro Kubota1) Kentaro Ueno1) Naoko Imuta2) Junichiro Nishi2) and Yoshifumi Kawano1)
1)Department of Pediatrics, Kagoshima University Hospital
2)Department of Microbiology, Kagoshima University Graduate School of Medical and Dental Sciences
Abstract
Cronobacter sakazakii is a gram-negative bacillus widely found in the natural environment. We report a case of a very low-birth-weight infant with congenital heart disease who had ventilator-associated pneumonia with bacteremia caused by C. sakazakii. The boy was born at 32 weeks and 1 day of pregnancy and weighed 1,154 g. Ventilator, peripherally inserted central venous catheter (PI), and arterial lines were required for the management of interrupted aortic arch type B complex. The infant developed ventilator-associated pneumonia 33 days after birth and was started on ampicillin and ceftazidime. As C. sakazakii was detected in the aspirated sputum and blood cultures, the antibiotics were changed to cephozoplan. On the following third day, though the pneumonia improved, the bacteremia persisted and thrombocytopenia was observed. On the fifth day, blood cultures continued to detect C. sakazakii and the PI and arterial lines were replaced. Amikacin was added on the sixth day. Subsequently, there was an increase in platelet count, and blood cultures became negative. Antimicrobial medication was administered for two weeks. The pathogen was not detected in the powdered infant formula milk. In cases of bacteremia from ventilator-associated pneumonia where there is difficulty in reinserting an indwelling catheter, the catheter should be removed promptly, especially if the causative organism is C. sakazakii.




Case Report
Title
Three Cases of Subgaleal Hematoma Due to Hair Pulling
Author
Kyosuke Ibi Katsuhiko Kitazawa Kohei Tonosono Mariko Arakawa Chika Uchiyama Yasuyuki Yamamoto Akihiro Iguchi Hironobu Kobayashi Masayoshi Senda and Akihito Honda
Department of Pediatrics, Asahi General Hospital
Abstract
Cases of subgaleal hematoma (SGH) during childhood have rarely been reported. Here, we report three cases of SGH in schoolgirls caused by hair pulling. Two of them were 6 years old, and the other was 11 years old. All patients presented to our emergency department with a chief complaint of scalp swelling, but with no contusions, hair loss, or neurological deficits. Computed tomography revealed no significant intracranial findings. The patients had no underlying diseases or coagulopathies, and they were not taking any medications. The hematomas were caused by hair pulling, in one case by a friend, and in the other two, by their mothers. In one of the cases, the hematoma was surgically aspirated because it worsened after hospitalization. The remaining two patients recovered conservatively. Neither had severe anemia nor needed blood transfusion. Further investigation into the family background of the two cases caused by the mothers revealed social problems in their parenting environments. Together with the public health nurses, we followed up with the family in one case. In the other case, the child guidance center decided to place the patient under temporary protective care due to history of repeated physical abuse by her parents. Conservative treatment suffices in most cases, but they are often associated with physical abuse. Thus, when a child presents with SGH, the social environment should be promptly evaluated.




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