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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.126, No.6, June 2022
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Original Article
Title
Children with Congenital Heart Disease and Home Medical Care Support
Author
Hirotaka Koga Jun Muneuchi Mamie Watanabe Yuichiro Sugitani Naoki Kawaguchi Ryohei Matsuoka Yuki Iwaya Shunichi Adachi and Yasuhiko Takahashi
Department of Pediatrics, Kyushu Hospital, Community Healthcare Organization
Abstract
Recent improvements in the survival of children with congenital heart disease (CHD) have increased the requirements of home medical care, but some children carry risks of life-threatening events or death. Between 2011 and 2018, we retrospectively reviewed 57 children with CHD who required two or more home medical care procedures, including oxygen supplementation, tracheostomy and mechanical ventilation, tube feeding, home nursing, and bedridden procedures. Age at the time of introduction of home medical care was 8 (0―188) months. There were 30 female children. During the follow-up period of 66 (16―405) months, there were 7 deaths, and 1.0 (0―6.2) unexpected hospitalizations per year. When we compared living and dead subjects, there were significant differences in body mass index (BMI) (14.5 [9.2―21.7] vs 11.9 [8.2―15.3] kg/cm2, P=0.022), plasma levels of brain natriuretic peptide (BNP) (30.8 [4.0―682.7] vs 227.0 [16.5―883.6] pg/dL, P=0.014), and Ross or NYHA functional classification class III or IV (60% vs 100%, P=0.038). A logistic regression analysis revealed that neither death nor unexpected hospitalization was associated with BMI, BNP levels, and severity of heart failure. In conclusion, BMI, plasma levels of BNP, and severity of heart failure were important risk factors in children with CHD requiring home medical care.
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Original Article
Title
Implementation of Behavioral Therapy Coaching for Childhood Obesity
Author
Katsuhiko Yamada and Miki Inutsuka
Department of Pediatrics, Sasebo Chuo Hospital
Abstract
We analyzed the effects of incorporating coaching into behavioral therapy for pediatric obesity on obesity index and hospital visit persistence. Thirty-four children (median age 10 years, male/female ratio 1:6) who visited our department between August 2010 and December 2016 were provided with a coaching session in which children and their parents could speak up independently in addition to conventional guidance. We collected data from medical records on the subjects' background, obesity index, duration of hospital visits, environmental factors, and interview records, for a treatment and observation period of 3 years from the first visit. Obesity index decreased continuously from the first visit to 3 months (p<0.0001), 6 months (p=0.0141), 9 months (p=0.0005), and 12 months (p=0.0023). There was a significant forward correlation between the duration of hospital visits and the decrease in obesity index (Rs=0.352, p=0.0447). Although the duration of hospital visits was shorter among children who refuse to go to school (p=0.0200), other environmental factors had a limited effect on the change in obesity index. The decrease in obesity index following the recording of the child's spontaneous comments toward behavioral change was larger (p=0.0010), while the decrease in obesity index following the recording of someone's actions that undermined the child's autonomy was smaller (p=0.0111), and often triggered the discontinuation of hospital visits. Behavioral therapy for childhood obesity via coaching might be effective in reducing obesity index in the medium term, with fewer dropouts from hospital visits.
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Original Article
Title
Thiopurine-induced Pancreatitis in Children with Inflammatory Bowel Disease
Author
Kirie Otsu Tatsuki Mizuochi Ryosuke Yasuda Ken Kato Yuko Shirahama Hirotaka Sakaguchi and Yushiro Yamashita
Department of Pediatrics and Child Health, Kurume University School of Medicine
Abstract
Background: Dose dependent side effects of thiopurine include leukopenia, alopecia, and liver dysfunction. Genotyping of NUDT15 codon 139 can predict these adverse events. However, dose-independent side effects such as fever, rash, joint pain, and pancreatitis have remained. The aim of this study was to determine clinical features of dose-independent side effects of thiopurine in children with inflammatory bowel disease (IBD).
Methods: Subjects were children under 16 years of age who were diagnosed with ulcerative colitis (UC) or Crohn's disease (CD) at Kurume University Hospital between 2011 and 2020. We retrospectively enrolled patients who received thiopurine.
Results: A total of 56 patients including 35 UC and 21 CD patients were enrolled. All patients received azathioprine. Four patients (7%) had thiopurine-induced pancreatitis, including 3 UC (9%) and 1 CD (5%). Median age was 8.5 years old. All patients had abdominal pain, including 2 with fever. All patients met the diagnostic criteria for acute pancreatitis, with elevated CRP (median, 10.6 mg/dL). Median period from initiation to onset was 21.5 days. Median dose of azathioprine at the time of onset was 0.9 mg/kg/day.
Conclusions: Incidence of thiopurine-induced pancreatitis in pediatric IBD was 7%. The onset period was between 2 and 4 weeks after initiation of thiopurine. All patients had acute pancreatitis with elevated CRP, some also had fever.
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Case Report
Title
Exacerbation of Cardiac Dysfunction after Bacterial Infection 12 Years Following Hematopoietic Cell Transplantation against Acute Lymphoblastic Leukemia: A Case Report
Author
Kengo Moriyama1)5) Ai Yoshimi1) Keisuke Kato1) Chie Kobayashi2)6) Junko Shiono3) Takashi Murakami3)6) Kazutoshi Koike1) Hitoshi Horigome3)6) Yoshiro Chiba4) Minoru Murata4) and Masahiro Tsuchida1)
1)Division of Pediatric Hematology and Oncology, Ibaraki Children's Hospital
2)Division of General Pediatrics, Ibaraki Children's Hospital
3)Division of Pediatric Cardiology, Ibaraki Children's Hospital
4)Department of Cardiology, Mito Saiseikai General Hospital
5)Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University
6)Department of Child Health, Faculty of Medicine, University of Tsukuba
Abstract
Anthracycline-induced cardiotoxicity is one of the most significant late adverse effects in childhood oncology, characterized by chronic progressive nature and increased risk in a dose-dependent manner. We encountered a 29-year-old woman who exhibited exacerbation of cardiac dysfunction related to anthracycline-induced cardiotoxicity following bacterial bronchial pneumonia. The patient was diagnosed with lymphoma leukemia syndrome at 13 years of age, and went into remission with chemotherapy of mature B-cell lymphoma. Twenty-two months following the initial diagnosis, the patient developed bone marrow recurrence and was diagnosed with acute lymphoblastic leukemia (ALL) for the first time. Although chemotherapy for ALL gave rise to the second remission, she developed a second recurrence just before transplantation. She received tandem autologous and allogeneic hematopoietic cell transplantation each after preconditioning, including 12 grays of total body irradiation at age 16, inducing long-term remission. The cumulative dose of anthracyclines was 516 mg/m2 of doxorubicin equivalent. Twelve years after the last transplant, the patient was admitted to our hospital brochopneumonia due to Haemophilus influenzae, and two days after admission, she presented with shock and multiple organ failure. Although she met the diagnostic criteria for septic shock, the main contributing factor was exacerbating anthracycline-induced cardiac dysfunction triggered by the infection. The patient required intensive care, including ventilatory management and peritoneal dialysis. Even after recovery, chronic cardiac dysfunction continued so that she could not be weaned from dependence on peritoneal dialysis. The patient required ventricular resynchronization therapy. Long-term observation is necessary for anthracycline-induced cardiac dysfunction.
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Case Report
Title
A Girl Who Developed Drug-induced Renal Dysfunction after Intravenous Acyclovir Administration
Author
Haruka Yamamura Mari Okada Emi Kijima Yousuke Ichigi Hisae Nakatani Haruna Yokoyama Masako Imai Natsuko Suzuki Masayuki Nagasawa and Akihiro Oshiba
Department of Pediatrics, Musashino Red Cross Hospital
Abstract
Drug-induced nephrotoxicity is a major side effect of the antiviral drug, acyclovir. Generally, it occurs in elderly people with renal impairment, and children are less likely to develop acute kidney injury (AKI) following acyclovir exposure. The main mechanism of renal dysfunction associated with acyclovir involves renal failure due to crystal precipitation in the renal tubules, but may also be due to acute tubulointerstitial nephritis (ATIN). Here, we report a 9-year-old girl with clinically mild encephalitis/encephalopathy with a reversible splenial lesion who developed drug-induced renal dysfunction after intravenous acyclovir administration. She developed stomachache, dry cough, fever, edema, and eruptions during acyclovir administration, and laboratory tests showed elevated serum creatinine level, pyuria, glycosuria, and crystalluria. A diagnosis of drug-induced crystal nephropathy and ATIN was made. Although, the efficacy of steroid treatment for drug-induced ATIN is controversial, a previous report suggested that immediate steroid therapy reduced the risk of chronic kidney disease. In reference to this report, we administered high-dose methylprednisolone, and the patient's serum creatinine level improved to the baseline approximately 5 months later. Acyclovir-induced nephrotoxicity usually lacks pathognomonic symptoms, such as oliguria. Therefore, it should be suspected based on nonspecific symptoms, such as fever or stomachache, and blood and urine tests should be performed. In conclusion, pediatricians should be aware of the need for early detection of acyclovir-induced nephrotoxicity to protect children's kidney function.
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Case Report
Title
Brain Abscesses with Fusobacterium nucleatum Established by 16S Ribosomal RNA Gene Sequencing from Cerebrospinal Fluid
Author
Haruka Yuki1) Kosuke Kohashi1) Takashi Kikuchi2) Yoshihiko Horimoto1) Shunsuke Shinozuka1) Hiroshi Okada1) Kazuhiro Suzuki1) Masato Mori1) and Ryugo Hiramoto1)
1)Department of Pediatrics, Matsudo City General Hospital, Children's Medical Centre
2)Division of Bacteriology, Chiba Prefectural Institute of Public Health
Abstract
We report a first pediatric case of brain abscesses caused by Fusobacterium nucleatum which was diagnosed by 16S ribosomal RNA (rRNA) gene sequencing from cerebrospinal fluid (CSF). 16S rRNA gene sequencing can identify bacteria from pus, when blood culture and CSF culture are negative.
A 1-year-old boy with enamel dysplasia and advanced caries was taken to a clinic with a history of fever and vomiting for a week. He was diagnosed with bacterial meningitis, and antibiotics were administered. However, left abducens nerve palsy and torticollis appeared. Brain computed tomography showed mass lesions, and he was transferred to our hospital. Gadolinium-based magnetic resonance imaging showed mass lesions with ring-shaped enhancement. Blood and CSF cultures were negative, but brain abscesses were considered to be the most likely diagnosis. Direct sampling for diagnosis was considered challenging because the lesion was deep. The fever subsided and the mass lesions shrank as antibiotics were continued for eight weeks. After completion of treatment, 16S rRNA gene sequencing detected Fusobacterium nucleatum in the CSF, confirming the diagnosis of brain abscess. Fusobacterium nucleatum, which is endemic to the oral cavity, was thought to have spread hematogenously from caries associated with enamel dysplasia.
CSF analysis by 16S rRNA gene sequencing can identify causative bacteria and confirm a diagnosis in cases in which direct sampling of abscesses are challenging.
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Case Report
Title
Acute Myopericarditis after COVID-19 Vaccination
Author
Naoki Yogo1) Toshihiko Nonaka1) Kei Honda1) Nozomi Nasu2) Yuichiro Muto1) and Katsuki Hirai1)
1)Department of Pediatrics, Japanese Red Cross Kumamoto Hospital
2)Department of Pediatrics, Kumamoto Regional Medical Center
Abstract
With the increase in the number of people receiving coronavirus disease 2019 (COVID-19) vaccines, reports of myocarditis have increased. However, there are currently no case reports on this matter in Japan. We report a case of a 12-year-old boy who developed myopericarditis after COVID-19 vaccination. Two days after receiving the vaccine, he experienced chest discomfort. Due to development of severe chest pain and elevated troponin levels, noted 3 days after his vaccination, he was transferred to our hospital. Based on the examination, he was diagnosed with myopericarditis. His symptoms improved after hospitalization. He was discharged on the seventh day. Myocarditis should be considered in patients presenting with chest pain after mRNA vaccination.
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Case Report
Title
Cerebral Venous Thrombosis in a 14-year-old with Crohn's Disease
Author
Shogo Horikawa1) Jiro Inagaki2) Yusuke Ono1) Tetuzi Sato2) Kenichi Takano1) Junzi Kamizono1)2) and Masano Amamoto1)
1)Children's Medical Center, Kitakyushu City Yahata Hospital,
2)Department of Pediatric Hematology/Oncology, Kitakyushu City Yahata Hospital
Abstract
Inflammatory bowel disease (IBD) is associated with a high risk of venous thromboembolism, especially deep-vein thrombosis of the lower extremities, pulmonary embolism, and portal and mesenteric vein thrombosis. Here, we report a case of a child who developed cerebral venous sinus thrombosis in the course of Crohn's disease (CD). A 14-year-old boy was diagnosed with small and large intestine type CD upon close examination of abdominal pain, chronic diarrhea, and marked weight loss. He was initially treated with oral prednisolone, 5-ASA, component nutrition, and improvement of PCDAI (47.5 points to 5 points) and remission of intestinal lesions were confirmed. Twenty-eight days later, he complained of pulsatile headache, without accompanying symptoms such as vomiting, elevated blood pressure, or intraocular pressure. As a result of head CT and MRI, a diagnosis of cerebral venous sinus thrombosis of the right transverse sinus to the sigmoid sinus was made. Blood coagulation tests at the time of thrombosis onset did not detect any genetic or acquired factors predisposing to thrombosis. One month later, imaging studies confirmed the disappearance of thrombus at the lesion site and the reopening of blood flow. If intracranial hypertension occurs during treatment of inflammatory bowel disease, cerebral venous sinus thrombosis should be suspected, and appropriate imaging and therapeutic intervention should be sought.
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