Journal of Yeungnam Medical Science

Case report

Hematology
Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report: Juhyung Kim, Soyoon Hwang, Narae Hwang, Yeonji Lee, Hee Jeong Cho, Joon Ho Moon, Sang Kyun Sohn, Dong Won Baek; J Yeungnam Med Sci. 2023;40(3):283-288. Published online July 28, 2022; DOI: https://doi.org/10.12701/jyms.2022.00353

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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.

Citations

Citations to this article as recorded by

Zahnmedizinische Betreuung seltener Erkrankungen: Fallberichte zu hereditärer sensorischer und autonomer Neuropathie sowie zu schwerer chronischer Neutropenie
Wieland Elger
Kinder- und Jugendmedizin.2025; 25(03): 162. CrossRef
Severe Congenital Neutropenia in a Newborn Caused by a Novel Mutation in the ELANE Gene—First Report From North Macedonia and a Literature Review: A Case Report
Nikolina Zdraveska, Teodora Trajkovska, Aleksandra Jovanovska, Arjeta Hasani, Mica Kimovska Hristova, Vjekoslav Krzelj
Case Reports in Pediatrics.2025;[Epub] CrossRef
ПЕРВИННІ МІНОРНІ ІМУНОДЕФІЦИТИ ЯК ПРИЧИНА РОЗВИТКУ ІМУНОЗАЛЕЖНОЇ ПАТОЛОГІЇ У ЛЮДЕЙ: ЕТІОЛОГІЯ, ЕПІДЕМІОЛОГІЯ, КЛАСИФІКАЦІЯ, ДІАГНОСТИКА І ЛІКУВАННЯ (СИСТЕМАТИЧНИЙ ОГЛЯД)
Dmytro Maltsev
Immunology and Allergology: Science and Practice.2024; (3-4): 37. CrossRef

Original article

Infectious Disease
Does oral doxycycline treatment affect eradication of urine vancomycin-resistant Enterococcus? A tertiary hospital study: Yoonjung Kim, Sohyun Bae, Soyoon Hwang, Ki Tae Kwon, Hyun-Ha Chang, Su-Jeong Kim, Han-Ki Park, Jong-Myung Lee, Shin-Woo Kim; Yeungnam Univ J Med. 2020;37(2):112-121. Published online February 18, 2020; DOI: https://doi.org/10.12701/yujm.2019.00430

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Abstract PDF Supplementary Material

Background
Vancomycin-resistant Enterococcus (VRE) has become more common in nosocomial infections, especially in urine samples. However, until now, no treatment regimen has been proven to effectively eradicate urine VRE colonization. Therefore, to evaluate the efficacy of doxycycline in eradicating urine VRE and shortening VRE isolation period, we compared VRE colony detection period between doxycycline-treated and untreated patients.
Methods
A retrospective cohort study of 83 patients with VRE colonization in urine cultures was conducted at a tertiary academic hospital from January 2011 to February 2018. Kaplan-Meier survival analysis was used to evaluate eradication rates in the treatment and non-treatment groups. Factors affecting urine VRE colonization persistence were analyzed by multiple logistic regression analysis.
Results
The overall rate of VRE eradication during the entire hospital stay was higher in the doxycycline treatment group (90.5%) than in the non-treatment group (58.1%, p=0.014). Survival analysis showed that the 5-, 10-, and 20-day cumulative eradication rates were 78.3%, 100%, and 100% in the doxycycline treatment group, and 18.5%, 45.7%, and 67.8% in the non-treatment group, respectively, thereby indicating that eradication rates were higher in the doxycycline treatment group than in the non-treatment group (p<0.001). Only doxycycline treatment was shown to affect urine VRE colonization persistence in multivariate logistic regression analysis
Conclusion
Doxycycline treatment enhanced the eradication rate of urine VRE colonization and appeared to be useful in shortening VRE isolation period.

Citations

Citations to this article as recorded by

Therapeutics for Vancomycin-Resistant Enterococcal Bloodstream Infections
Kelly A. Cairns, Andrew A. Udy, Trisha N. Peel, Iain J. Abbott, Michael J. Dooley, Anton Y. Peleg
Clinical Microbiology Reviews.2023;[Epub] CrossRef
Aminopenicillins for treatment of ampicillin-resistant enterococcal urinary tract infections
Kristen Bunnell, Amy Duong, Thomas Ringsred, Asia Mian, Sanaya Bhathena
American Journal of Health-System Pharmacy.2022; 79(13): 1056. CrossRef
Doxycycline for ESBL-E Cystitis
Tomasz Jodlowski, Charles R Ashby, Sarath G Nath
Clinical Infectious Diseases.2021; 73(1): e274. CrossRef

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