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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.122, No.7, July 2018
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Review
Title
Neurological Examinations for Functional Movement Disorders
Author
Mariko Y Momoi
Emeritus Professor, Pediatrics, Jichi Medical University
Ryomo Seishiryogoen
Abstract
Here we describe a general approach to the assessment of patients with functional neurologic disorders. Functional movement disorder is a common clinical feature of functional neurologic disorders, of which prevalence in neurologic clinics is relatively high. However, the diagnosis of functional movement disorders has been challenging because of the lack of cognition and interest among physicians, including neurologists. The recent diagnosis of this condition relies on positive clinical signs rather than the exclusion of organic disorders. Systematic clinical examination of the patients based on an understanding of the unique neurological features and signs is of great help for the early and precise diagnosis of this condition. The appropriate interpretation of the positive signs and diagnosis of patients at the early stage of the illness can improve the patients' distorted concept of their disease, which is critical for good prognosis.
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Original Article
Title
Congenital Heart Disease with Intracranial Hemorrhage in the Neonatal Period
Author
Masahiro Tsubura1) Tomoaki Murakami2) and Koji Higashi2)
1)Division of Neonatology, Chiba Children's Hospital
2)Division of Cardiology, Chiba Children's Hospital
Abstract
Background: The survival of patients with congenital heart disease (CHD) has dramatically improved. Therefore, neurodevelopmental outcomes in survivors are receiving much attention. We investigated intracranial hemorrhage (ICH), which could have a big impact on the neurodevelopmental outcomes, in neonates with CHD.
Methods: We retrospectively reviewed the medical records and images in 793 neonates at our institute between January 2009 and January 2014.
Results: The incidence of ICH was 57 of 793 (7%) patients and 18 of 241 (7%) patients with CHD, compared to 39 of 552 (7%) patients with non-CHD. In analysis of CHD patients apart from extremely low birth weight infants and very low birth weight infants, the incidence of ICH was 14 of 232 (6%) patients with CHD and 10 of 463 (2%) patients with non-CHD (p<0.01). Among ICH-CHD patients, 6 cases died in early infancy, and 5 of 8 survivors suffered from neurodevelopmental problems. Although there was no significant difference in the incident of ICH between pre and postnatally diagnosed CHD patients, all 6 ICH patients with CHD who were not prenatally diagnosed died.
Conclusions: The prognosis of the ICH in patients with CHD is poor. Advances in perinatal, neonatal and perioperative care based on prenatal diagnosis may reduce the prevalence of the ICH and related neurodevelopmental morbidities.
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Original Article
Title
Clinical Analysis of a Small Outbreak of Community-transmitted Oseltamivir-resistant Influenza A (H1N1) pdm09 in a Pediatric Outpatient Clinic
Author
Shuji Nakata
Nakata Pediatric Clinic
Abstract
A small outbreak of community-transmitted oseltamivir-resistant influenza A (H1N1) pdm09 was identified in Sapporo, Hokkaido, between December 2013 and February 2014. Out of 104 oseltamivir-resistant A (H1N1) pdm09 influenza viruses detected in Japan during the 2013/2014 influenza season, 34 were from the patients in Sapporo. Thirty-three of them were not treated with neuraminidase inhibitors (NIs) before the influenza specimen collection. The viruses detected from them were genetically closely related to each other, suggesting the spread of a single variant from one patient to another.
The clinical data were compared among 9, 11 and 12 patients infected with oseltamivir-resistant influenza A (H1N1) pdm09, oseltamivir-susceptible influenza A (H1N1) pdm09 and influenza A (H3N2), respectively, in a pediatric outpatient clinic in Sapporo. The median duration of the fever in the 3 groups was 2, 3 and 3 days, respectively, and median maximum body temperature was 39.2, 39.4 and 39.3°C, respectively, suggesting no significant difference among them. The median duration of the fever was shorter in 28 NIs-treated cases (2 days) than in 4 nontreated cases (4 days). In NIs-treated cases, there was no difference in the period of fever between 16 oseltamivir-treated cases (2 days) and 12 zanamivir-treated cases (2 days).
Our data suggested that the difference of influenza A subtype and antiviral resistance was not associated with the duration of fever. Although it was not statistically analyzed, the effectiveness of oseltamivir could be equal to that of zanamivir regarding infection with oseltamivir-resistant viruses.
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Original Article
Title
Epidemiological Survey of Critically Ill Pediatric Patients Requiring Intensive Care
Author
Takeshi Hatachi1) Masayo Tsuda2) Miyako Kyogoku1) Shingo Adachi3) Keisuke Sugimoto4) Kazue Moon1) Kanako Isaka1) Yu Inata1) Yoshiyuki Shimizu1) and Muneyuki Takeuchi1)
1)Department of Intensive Care Medicine, Osaka Women's and Children's Hospital
2)Division of Pediatric Hospital Medicine, Hyogo Prefectural Kobe Children's Hospital
3)Senshu Trauma and Critical Care Center, Rinku General Medical Center
4)Department of Pediatrics, Kindai University Faculty of Medicine
Abstract
We prospectively conducted an observational study of critically ill pediatric emergency patients aged <15 years in three medical regions in the southern part of Osaka Prefecture for 1 year, beginning in April 2015. We identified 196 patients with a median age of 2 years. Approximately 41% of the patients had underlying diseases. Of these, 87% demonstrated endogenous diseases, 44% had infections, and 10% suffered injuries secondary to suspected child abuse. The 1-year incidence of critically ill pediatric emergency patients was 0.641 per 1,000 children, the mortality rate was 10%, and 19% died or had decreased neurological function on evaluation. Cardiopulmonary resuscitation, impaired consciousness after 24 h, acute virus-associated encephalopathy, and mechanical ventilation were associated with poor outcomes.
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Case Report
Title
Severe Respiratory Suppression Due to Dihydrocodeine Intoxication in a Neonate
Author
Tatsuki Kondo1) Tetsuya Fukuoka1) Tsutomu Shioda1) Mika Sakuma1) Kenji Komatsu1) Tatsuki Ota1) Megumi Sato1) Yumiko Okubo1) and Toshihiro Tanaka2)
1)Pediatric Department, Shizuoka Saiseikai General Hospital
2)Pediatric Department, Shizuoka Welfare Hospital
Abstract
A 23-day-old boy who was given 6 mg dihydrocodeine for 2 days as an antitussive developed severe respiratory suppression and was transferred to our hospital. He had remarkable cyanosis due to frequent apnea, and required mechanical ventilation. White blood cell count, C-reactive protein level, and the number of spinal fluid cells were within the normal range. Head computed tomography, chest radiography, and echocardiography also yielded normal results. Meningitis, sepsis, heart disease, and intracranial hemorrhage were not suspected. He was extubated on the 6th day and was discharged on the 17th day. Blood concentrations of dihydrocodeine and dihydromorphine at the time of admission were about 10 times higher than normal values. His genotype with respect to CYP2D6, which is involved in the metabolism of dihydrocodeine to dihydromorphine, was CYP2D6＊1/＊10-＊36. Hence, his metabolic capacity for dihydrocodeine was considered normal. The respiratory suppression was attributed to increased levels of dihydrocodeine and dihydromorphine in the blood. Dihydrocodeine intoxication in children is likely to be lethal, even when the drug is used appropriately. It must be recognized that the use of dihydrocodeine in children carries a high risk.
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Case Report
Title
A Case of Stevens-Johnson Syndrome with Respiratory Failure Caused by Acetaminophen
Author
Hikaru Sunakawa Junko Yamanaka Mayu Koto Yuri Yoshimoto Mizue Tanaka Yoshiaki Okuma Hideko Uryu Noriko Sato and Hiroyuki Shichino
Department of Pediatrics, National Center for Global Health and Medicine
Abstract
We report a 7-year-old girl who was given a diagnosis of Stevens-Johnson syndrome (SJS) with respiratory failure caused by acetaminophen and subsequently was complicated by bronchiolitis obliterans (BO) in the chronic phase. She was hospitalized due to fever and extensive blisters with pain. We prescribed acetaminophen to deal with the high fever and pain. Three hours after the acetaminophen drip infusion she suffered severe respiratory distress and received intensive treatment. Initially we did not realize the fact of taking medication and did not suspect drug allergy. From the medication history interview, clinical course and positive result of drug lymphocyte stimulation test, we diagnosed SJS caused by acetaminophen. SJS improved using steroid drugs. Regarding her respiratory condition, wheezing persisted after the acute phase and obstructive ventilatory impairment continues. We suspect that BO was a complication caused by SJS. There is no effective treatment for BO at present but there are reports of severe BO patients receiving lung transplantation. The obstructive ventilatory impairment in the present case is deteriorating recently despite alternative treatments. In the future she might need to receive lung transplantation. As acetaminophen is a common antipyretic and pain reliever for children, we must be aware of the severely adverse reactions that can occur.
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Case Report
Title
Early Surgical Intervention for Loeys-Dietz Syndrome in a 15-year-old Boy
Author
Naofumi Hanyu1) Yasuyuki Morishima1)2) Masaru Shimura1) Wakako Suda1) Shinji Suzuki1) Souken Go1) Shigeo Nishimata1) Yasuyo Kashiwagi1) Hironao Numabe1)2) and Hisashi Kawashima1)
1)Department of Pediatrics, Tokyo Medical University
2)Department of Clinical Genetics Center, Tokyo Medical University
Abstract
We report a case of a 15-year-old boy with Loeys-Dietz syndrome (LDS). Because of pectus excavatum and scoliosis, the patient was referred to our hospital for further investigations. On general physical examination, he had hypertelorism, broad uvula, long fingers and toes, pes planus, and aortic root dilatation (41 mm) found by echocardiogram. We diagnosed LDS based on the mutation in TGFBR2 and his phenotypes. He was followed up and prescribed β-blocker and angiotensin II receptor blockers for blood pressure control. After 10 months, to prevent the risk of aortic dissection due to aortic root expansion, we performed valve-sparing aortic root replacement (David Procedure). LDS is a connective-tissue disorder similar to Marfan syndrome (MFS), however aortic dissection may occur even in early childhood with LDS unlike MFS. It is important for pediatricians to recognize the syndrome as early as possible.
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Case Report
Title
Acute Kidney Injury Induced by Acute Gastroenteritis in Two Adolescents with Renal Hypouricemia
Author
Maki Shimizu Masahiro Umeda Shuhei Onishi Satoshi Takehiro Tomoko Ichihara Hiromi Ohashi Shoko Fujii Zenichi Sakaguchi and Hiroko Kozan
Department of Pediatrics, Takamatsu Red Cross Hospital
Abstract
Exercise-induced acute kidney injury (AKI) is the main complication of renal hypouricemia. However, AKI with renal hypouricemia caused by factors other than exercise is extremely rare. We report AKI triggered by acute gastroenteritis in two cases with renal hypouricemia. Case 1 was a 15-year-old girl. Renal dysfunction was revealed when she suffered from diarrhea, nausea, vomiting and appetite loss persisting for a week. We diagnosed AKI with renal hypouricemia, because hypouricemia due to excessive urinary excretion of uric acid (UA) was confirmed, moreover serum UA and fractional excretion of UA (FEUA) were 1.0 mg/dL and 57.5%, respectively. Case 2 was a 14-year-old boy. Renal dysfunction was noticed when he suffered from fever, diarrhea, nausea, vomiting and appetite loss persisting for 5 days. We also diagnosed AKI with renal hypouricemia because his serum UA was 0.3 mg/dL and FEUA was 124%. Genetic analysis of GLUT9 (SLC2A9, which encodes glucose transporter 9) gene revealed 4 heterozygous missense mutations in case 2. Both adolescents did not have episodes of anaerobic exercise before the onset of AKI. It was speculated that AKI resulted from increase of oxidative stress and reduction of renal blood flow induced by gastroenteritis superimposed on a relative shortage of antioxidative capacity due to hypouricemia. There is a possibility of developing AKI with renal hypouricemia triggered by gastroenteritis, even in the absence of anaerobic exercise.
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Case Report
Title
Bilateral Hip Joint Dislocation Diagnosed as a Result of Short Stature and Posture Abnormality after Independent Walking
Author
Mana Tanaka1) Shigeru Maruyama1) Masashi Suda1) and Keisuke Nagasaki1)2)
1)Department of Pediatrics, Niigata Prefectural Central Hospital
2)Department of Pediatrics, Niigata University Medical & Dental Hospital
Abstract
The number of patients with congenital hip dislocation has decreased drastically due to prevention activities. Recently, however, patients who are given diagnoses after independent walking are considered problematic. Since it is impossible to repair this condition with brace treatment after independent walking, early detection is important. We present a 29-month-old girl who was given a diagnosis of bilateral hip joint dislocation when she visited our hospital for examination due to short stature and abnormal posture. No abnormality had been noted in her past health examinations. We found her to have increased lordosis, diagnosed as bilateral congenital hip dislocation by X-ray, and started traction treatment under hospitalization. Since reposition by traction was ineffective, invasive treatment is currently underway. Because this case had only mild limitation of abduction and flexion and showed no laterality of hip joint excursion or skin wrinkles, no abnormality was noticed during her examination as an infant. In 2015, the Japanese Orthopedic Association and Japan Children's Orthopedic Association published recommended guidelines for infant hip joint health examinations. Our patient met the referral criteria for a second screening for female infants and pelvic position at birth. In areas where the initial examination is performed by only a pediatrician, female infants with pelvic position birth should be considered for referral for secondary screening.
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Case Report
Title
Repeated Paradoxical Reactions in a Patient with Meningeal and Intracranial Tuberculosis during Antituberculous Treatment with Corticosteroids
Author
Kosuke Noma Takehiko Doi Ryota Komori Yuta Eguchi Daichi Ono Risa Matsumura Shinji Mochizuki Satoshi Okada and Masao Kobayashi
Department of Pediatrics, Hiroshima University Hospital
Abstract
A 14-year-old boy who had developed a cerebellar tumor at the age of 10 years underwent surgery to remove the tumor at the age of 13 years. Nine months after surgery, he presented with fever and headache progressing to right sensorineural hearing loss, convulsions, apnea, and severe consciousness disturbance. Emergency ventricular drainage was performed to relieve the severe hydrocephalus. He was diagnosed with meningeal and intracranial tuberculosis based on the pathological findings and positive for tuberculosis polymerase chain reaction and Ziehl-Neelsen staining of cerebrospinal fluid. Antituberculous chemotherapy was initiated in combination with dexamethasone according to the appropriate guidelines, leading to gradual recovery from his clinical and laboratory manifestations. However, he had recurrent fever and headache associated with tapering dexamethasone. The paradoxical reaction of tuberculosis meningitis was considered by the results of cerebrospinal fluid examination and MRI imaging. Furthermore, cerebral artery stenosis detected by MRI was also presented as a paradoxical reaction during the tapering of prednisolone. Consequently, he has improved by continuation of antituberculous chemotherapy with the adjustment of the corticosteroid dosage although the right sensorineural hearing loss persists.
Thus, the paradoxical reaction might be clinically recognized during the treatment of central nervous system tuberculosis, suggesting the importance of careful observation and appropriate administration of corticosteroids.
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Case Report
Title
Use of Octreotide to Treat an Infant with Traumatic Chylothorax Thought to Have Been Caused by a Traffic Accident
Author
Ryoji Niino Yuko Kataoka Mitsuru Tsuge Satoshi Murata Reiji Miyawaki Hiroshi Ogasawara Koso Ueda and Yoichi Kondo
Department of Pediatrics, Matsuyama Red Cross Hospital
Abstract
Chylothorax in children is classified into traumatic, non-traumatic, and idiopathic, according to its cause. However, reports of traumatic chylothorax cases other than iatrogenic cases are extremely rare. We report our experience using octreotide to treat an infant with traumatic chylothorax thought to have been caused by a traffic accident. There are various causes for the occurrence of traumatic chylothorax in children, including traffic accidents, abuse, and falls. However, depending on the state of the injury, due consideration should be given to patients in whom the period from injury to onset is long and who exhibit no traumatic findings other than chylothorax. In our search of the literature, we found no cases in which octreotide was used to treat an infant with traumatic chylothorax. Octreotide is a drug that can be used relatively safely and could be a potential treatment option for children with traumatic chylothorax.
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