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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.124, No.7, July 2020
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Original Article
Title
Usefulness of Pathological Autopsy in Cases of Death among Patients with Neonatal Disease
Author
Atsushi Naitoh1)2) Atsushi Nemoto1) Mami Kobayashi1) Yohei Hasebe1) and Yuuki Maebayashi1)
1)Department of Neonatology, Perinatal Center, Yamanashi Prefectural Central Hospital
2)Department of Pediatrics, Faculty of Medicine, University of Yamanashi
Abstract
Autopsies are being performed at lower rates, including the neonatal field. We retrospectively investigated all deaths at the Perinatal Medical Center of Yamanashi Prefectural Central Hospital to examine the usefulness of pathological autopsy-based diagnosis in fatal cases of neonatal disease. Our analysis included 82 deaths among 2,861 neonatal patients admitted to the hospital since its establishment in September 2001 until September 2018. Patient and institutional characteristics were compared between early (2001-2009) and late periods (2010-2018). Then they were divided by an increase in the number of neonatal intensive care unit beds at the hospital to check for their effects on the rates of autopsies performed and positive findings. In addition, autopsy concordance with clinical findings was evaluated using the Goldman criteria. Autopsies were performed in 57.3% of deaths overall (57.3%). High autopsy rates were maintained up to recent years, that is, 62.5% (30/48) in the early period and 50.0% (17/34) in the late period. New findings were observed from pathological autopsies in approximately 29 cases (61.7%) and confirmed the diagnosis of major disease directly connected to the cause of death in 13 patients (27.7%). Pathological findings were also of great significance in cases where they corroborated the clinical diagnosis. They reinforced it and shed light on their clinical conditions. Pathological autopsy remains an important tool in determining patients' true causes of death today, despite the progress in various diagnostic technologies.
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Original Article
Title
A Study of Pediatric Patients with IgA Vasculitis in a Single Institution over a 10-Year Period
Author
Ayako Shiihashi Takahiro Momoi Akiko Koyama Akira Kojima Noritaka Huruya and Hajime Nishimoto
Department of Pediatrics, Saitama Citizens Medical Center
Abstract
Background:
IgA vasculitis is a common form of systemic vasculitis in children. Its clinical manifestations vary diversely, with some patients recovering only with rest and others requiring long-term treatment due to renal involvement and recurrence. We investigated 124 patients with IgA vasculitis in our hospital to elucidate its clinical features.
Methods:
We investigated the clinical course of 124 patients with IgA vasculitis, who were admitted to our hospital for the first time, between April 2009 and March 2019, and compared a group with nephritis to one without nephritis.
Results:
The median age was 5 years and the male-to-female ratio was almost equal. Four patients were hospitalized because of recurrence. The group with nephritis showed a higher proportion of children over 6 years of age than the group without nephritis. The recurrence and readmission rates due to recurrence were also higher in the group with nephritis. In addition, the group with nephritis showed a higher probability of development or relapse of abdominal pain after prednisolone (PSL) treatment and required longer periods of both hospitalization and PSL treatment.
Conclusion:
We found that being over 6 years of age, recurrence, and existence of abdominal pain resistant to PSL treatment, are risk factors for renal involvement in IgA vasculitis.
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Original Article
Title
Evaluation of the Efficacy of Manual Extraction under General Anesthesia for Refractory Constipation in Children
Author
Yuta Ariyama1) Nao Tachibana2) Chiho Nakamura2) and Kouji Murakoshi2)
1)Department of General Pediatrics, Tokyo Metropolitan Children's Medical Center
2)Department of Alimentary System, Tokyo Metropolitan Children's Medical Center
Abstract
Purpose: We evaluated the efficacy of manual extraction under general anesthesia for refractory constipation in children.
Methods: From March 1, 2010, to August 31, 2016, patients who had manual extraction under general anesthesia at Tokyo Metropolitan Children's Medical Center were included in this study. We excluded patients who used this technique as pretreatment for surgical operations. We gathered data by electronic medical records retrospectively. We defined patients as successes when they could spend 30 days without an additional technique like a cleansing enema, Gastrografin® enema, and manual extraction. Then, we evaluated the success ratio. We performed manual extraction with both a hand technique under general anesthesia and muscle relaxation by an anesthetist.
Results: We performed manual extraction under general anesthesia for 32 patients, and we excluded two patients. Therefore, we included 30 patients in this study. The success ratio of this technique for chronic functional constipation was 93% (14/15), the surgical disease was 40% (2/5), the physical disease was 33% (1/3), and psychiatry disease was 71% (5/7), respectively. No patient needed to extend the admission term because of a complication of this technique.
Conclusion: The success rate of manual extraction under general anesthesia for chronic functional constipation was 93%. We think that this technique is effective for refractory constipation in children, though the institutions where this technique can be performed are limited.
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Case Report
Title
A Case of Acute Rupture of the Mitral Chordae Tendineae in an Infant Following Fetal Exposure to Maternal Anti-SS-A Antibodies
Author
Risa Morita1) Kotaro Urayama1) Mitsunobu Sugino1) Masahiro Tahara1) Kazunori Yamada2) Eriko Okajima3) and Atsushi Ono3)
1)Department of Pediatrics, Tsuchiya General Hospital
2)Department of Cardiovascular Surgery, Tsuchiya General Hospital
3)Department of Pediatrics, Miyoshi Central Hospital
Abstract
Acute rupture of the mitral chordae tendineae in infants is a serious condition resulting in sudden onset of circulatory and respiratory disorders in previously healthy infants and requires emergency surgical treatment. Prodromal symptoms related to certain infectious diseases are seen in many cases; however, the etiology of the disease has not been clarified. The present case had prenatal exposure to maternal anti-SS-A antibodies, which might have damaged the papillary muscles, leading to rupture of the mitral chordae tendineae at 2 months of age. Emergency surgical repair of the mitral chorda and valve leaflet was successfully performed, and she is now doing well without any significant complications. As the incidence of death and neurological complications in such cases is considerably high, the condition must be diagnosed immediately and appropriately treated. Maternal anti-SS-A antibodies should be considered as the cause of the cardiac pathology if this condition occurs in early infancy without any prodromal symptoms.
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Case Report
Title
A Case Report of Trading Card Game-Induced Reflex Epilepsy
Author
Hisako Yamamoto1)2) Yusaku Miyamoto1)2) Kanako Takeda1)2) and Naoki Shimizu2)
1)Pediatric Department, Kawasaki Municipal Tama Hospital
2)Pediatric Department, St. Marianna University School of Medicine
Abstract
Reflex epilepsy is defined as seizures triggered by specific stimuli. It is divided into simple and complex groups. The complex group is also referred to as praxis-induced (PI) seizures. Here, we report a patient with card game-induced reflex epilepsy. He had three episodes of tonic-clonic convulsions and myoclonic movements that occurred while he was playing card games (Duel Masters®). We suspected that the convulsions were related to epilepsy; therefore, we performed an ictal (electroencephalogram) EEG. No seizures were captured on the EEG recording. However, a repetitive generalized spike and slow-wave on the EEG were observed only when the patient was engaged in card games. Because of this EEG finding and his history, we diagnosed him with card game-induced reflex epilepsy. The seizures did not appear when looking at a card, but only during card games. Hence, we assumed that the seizures were induced by decision making, which is a type of PI seizure. We failed to record the ictal EEG; however, epileptic discharges were only observed during the card game. Therefore, the myoclonic movement was considered to be a seizure. It suggested a relation between the PI seizure and idiopathic generalized epilepsy. Therefore, we recommend detailed medical interviews for the diagnosis of PI seizure.
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Case Report
Title
The Current State of Home Care Support of Infants at Our Hospital for Four Ambulatory Children with Complex Medical Conditions
Author
Yong Kye Lee1) Sayaka Terada1) Keiko Wada1) and Yuka Yotsumoto2)
1)Department of Pediatrics and Rehabilitation Medicine, Aijinkai Rehabilitation Hospital
2)Department of Pediatrics, Takatsuki General Hospital
Abstract
We report the current state of our hospital for the home care support of infants regarding four ambulatory children with complex medical conditions who require tracheostomy management. The underlying diseases are congenital central hypoventilation syndrome (2 cases), coloboma, heart defects, atresia choanae (also known as choanal atresia), growth retardation, genital abnormalities, and ear abnormalities (CHARGE) syndrome with acquired laryngeal atresia (1 case), and subglottic stenosis (a preterm infant). Each child was admitted to the hospital between the ages of 1 and 5 years for reasons such as caregiver's illness and substantial care burden on the parents. A childcare program involving nursery teachers was established in the hospital in the wake of self-mutilation associated with an autism spectrum disorder. We assigned nurses to work adjustments for early work and late work to control the high-risk behavior of the children. During the day, staff monitored the children and supported them through rehabilitation aimed at improving activities of daily living and promoting development. Three patients, who had to be hospitalized for a long time, moved back home after completing overnight stays at home and discussions in predischarge conferences. While continuing to provide home care support through planned short-term hospitalization, information sharing, and support to the local community were promoted, so that the patients could have a stable home life in the community.
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Case Report
Title
Two Cases of HIV Infection Where Pulmonary Biopsy Was Useful for Distinguishing between Tuberculosis and Lymphocytic Interstitial Pneumonia
Author
Mami Shimada Mizue Tanaka Tomomi Ota Yukari Atsumi Mari Honda Yuri Yoshimoto Yoshiaki Okuma Masao Kaneshige Hideko Uryu Junko Yamanaka Ayumi Mizukami Keiji Goishi Noriko Sato and Hiroyuki Shichino
Department of Pediatrics, National Center for Global Health and Medicine
Abstract
When introducing combination antiretroviral therapy (cART) for HIV infection, the treatment regimen differs depending on the presence or absence of opportunistic infections. Therefore, the diagnosis of complications before treatment is important but may be difficult if it is based on only the clinical picture or imaging findings. Case 1 was a 4-year-old boy with prolonged fever and wheezing who was diagnosed as having HIV infection. Clinical features and imaging findings suggested coexisting respiratory complications, but the results of some tests did not lead to a definitive diagnosis. It was difficult to distinguish between tuberculosis (TB) and lymphocytic interstitial pneumonia (LIP). So, we performed a thoracotomy for lung biopsy, which led to the diagnosis of LIP. Case 2 was a 4-year-old girl whose father was diagnosed with AIDS. The screening revealed that the infant had HIV infection. She was suspected of having miliary tuberculosis, but her acid-fast bacteria smear was negative, and it was difficult to distinguish between TB and LIP. We then performed a thoracoscopic lung biopsy, which led to the diagnosis of LIP. In both cases, cART was started after diagnosis, and both patients improved. TB and LIP have many similarities, and pulmonary biopsy is indispensable for a definitive diagnosis of LIP.
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Case Report
Title
Urinary Tract Infection Caused by Actinotignum schaalii in a 2-Year Old Boy
Author
Eiki Ogawa1)3) Toru Higuchi1) Takumi Shiohara1) Naoko Takahashi2) and Kenta Ito1)
1)Division of General Pediatrics, Aichi Children's Health and Medical Center
2)Clinical Laboratory, Aichi Children's Health and Medical Center
3)Division of Infectious Diseases, National Center for Child Health and Development
Abstract
Gram-positive rods (GPR) rarely cause urinary tract infections (UTI) and are often considered as contaminants when recovered from urine cultures. Actinotignum schaalii is a facultative anaerobic GPR. With the increased use of mass spectrograph analyzers, A. schaalii has been reported as an emerging cause of UTI in adults. We report a case of UTI caused by A. schaalii with a previously undiagnosed bladder diverticulum. A previously healthy 2-year old boy visited our emergency department with fever and lower abdominal pain. Pyuria was observed on day 2 of the illness, and abdominal ultrasonography revealed bladder diverticulum. A gram stain of urine specimen showed GPR, and we initiated ampicillin/sulbactam for presumed infection caused by anaerobic bacteria. On day 3 of the illness, his fever abated, and his general condition gradually improved. Mass spectrometry of GPR confirmed A. schaalii. Antibiotics were switched to amoxicillin/clavulanate, and he was treated for 10 days in total. Eight cases of A. schaalii infection in children have been reported. The diagnoses in seven of these cases were UTI, and all cases included urological underlying diseases. In conclusion, some GPR might be causative organisms of UTI in children with urological underlying diseases.
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Case Report
Title
Role of Abdominal Ultrasonography in Protein-Losing Gastroenteropathy with Gastrointestinal Cytomegalovirus Infection
Author
Yumi B. Tamura Tomoki Satou Chinami Matsumoto Shintaro Funaki Masaki Shibanuma Hiroki Izumo Shuji Yoshino Yuuji Fujii Hiroyuki Shimozono Keita Matsubara and Rika Okano
Department of Pediatrics, Hiroshima City Funairi Citizens Hospital
Abstract
Protein-losing gastroenteropathy (PLE), a condition associated with multifactorial etiology, is characterized by loss of protein through the gastrointestinal tract causing hypoalbuminemia and hypoproteinemia. The clinical presentation of PLE varies from mild edema to ascites depending on the underlying cause. Accurate identification of the cause is important because prognosis and treatment vary depending on the cause. Using abdominal ultrasonography, we serially evaluated gastric wall thickening and blood flow in a patient with PLE with intestinal cytomegalovirus (CMV) infection. Case: A 2-year-old boy was admitted with systemic edema, hypoproteinemia, and hypoalbuminemia. Protein leakage from the stomach was confirmed by (99m) Tc-labelled human serum albumin scintigraphy, and a polymerase reaction assay revealed CMV-DNA in the gastric mucosa, thereby confirming the diagnosis of PLE with CMV infection. Abdominal ultrasonography showed gastric wall thickening and increased blood flow in the gastric wall and the small intestine. Concurrent improvement was observed in clinical symptoms, serum albumin levels, and ultrasonography findings. The earliest improvement observed on ultrasonography was increased blood flow-related signals in the small intestinal wall. However, gastric wall thickening persisted. Upper gastrointestinal endoscopy revealed slight erythema and edema of the gastric mucosa, which was significantly correlated with ultrasonographic findings. Abdominal ultrasonography is useful to determine the disease status of PLE in patients with CMV infection.
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