Severe chronic neutropenia is classified as severe congenital, cyclic, autoimmune, or idiopathic. However, there is a lot of uncertainty regarding the diagnosis of severe congenital neutropenia (SCN) and chronic idiopathic neutropenia, and this uncertainty affects further evaluations and treatments. A 20-year-old man presented with fever and knee abrasions after a bicycle accident. On admission, his initial absolute neutrophil count (ANC) was 30/µL. He had no medical history of persistent severe neutropenia with periodic oscillation of ANC. Although his fever resolved after appropriate antibiotic therapy, ANC remained at 80/µL. Bone marrow (BM) aspiration and biopsy were performed, and a BM smear showed myeloid maturation arrest. Moreover, genetic mutation test results showed a heterozygous missense variant in exon 4 of the neutrophil elastase ELANE: c597+1G>C (pV190-F199del). The patient was diagnosed with SCN. After discharge, we routinely checked his ANC level and monitored any signs of infection with minimum use of granulocyte colony-stimulating factor (G-CSF), considering its potential risk of leukemic transformation. Considering that SCN can be fatal, timely diagnosis and appropriate management with G-CSF are essential. We report the case of a patient with SCN caused by ELANE mutation who had atypical clinical manifestations. For a more accurate diagnosis and treatment of severe chronic neutropenia, further studies are needed to elucidate the various clinical features of ELANE.
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http://dx.doi.org/10.12701/jyms.2022.00353 | DOI Listing |
Pediatr Res
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Center for Genetic Medicine, Children's National Research Institute, Washington, DC, USA.
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January 2025
Pediatrics, The University of Tennessee Health Science Center, Memphis, Tennessee, USA.
A term, healthy infant presented with respiratory distress and severe pulmonary hypertension (PH). With an unclear aetiology and the intent to decrease right ventricular afterload, pulmonary vasodilators were initiated. Follow-up imaging revealed a supravalvular mitral ring as the cause of the PH which resolved after surgical resection of the membrane.
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December 2024
Langford Vets Small Animal Referral Hospital, University of Bristol, Langford, Bristol, BS40 5DU, United Kingdom; Eastcott Referrals, Edison Park, Swindon, SN3 3FR, United Kingdom.
Introduction: Severity of aortic stenosis (AS) in humans is classified using a staging system based on two-dimensional echocardiographic changes, which considers the extent of global cardiac damage. Currently, classification of canine AS is based on trans-aortic pressure gradient (PG) alone. This study aimed to retrospectively classify dogs with AS based on an adapted human staging system, exploring feasibility of classification and the association between stage and features such as PG and clinical signs.
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December 2024
Facultad de Medicina, Universidad Autónoma de Campeche, Campeche.
Background: Congenital neutropenia is a primary immunodeficiency characterized by quantitative anomalies in neutrophil counts. It is classified as mild, moderate, or severe. Hematopoietic stem cell transplantation stands as a potential therapeutic intervention; nevertheless, graft-versus-host disease emerges as a main complication.
View Article and Find Full Text PDFRev Alerg Mex
December 2024
Departamento de Inmunología, Hospital Infantil de Especialidades de Chihuahua; Facultad de Medicina y Ciencias Biomédicas, Universidad Autónoma de Chihuahua.
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