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Acute Chest Syndrome in Sickle Cell Disease: Clinical Presentation and Course
Open accessAbstract
Acute chest syndrome (ACS) is an important cause of morbidity and mortality in sickle cell disease (SCD). Previous studies reported conflicting pictures of ACS making therapeutic interventions difficult. The Cooperative Study of Sickle Cell Disease prospectively followed 3,751 patients enrolled from birth to 66 years of age for ACS. Data on presenting signs and symptoms, laboratory findings, and hospital course were collected. There were 1,722 ACS episodes in 939 patients. Young children (age 2 to 4 years) presented with fever and cough, a negative physical exam, and rarely had pain. Adults were often afebrile and complained of shortness of breath, chills, and severe pain. Upper lobe disease was more common in children; multilobe and lower lobe disease affected adults more often. Severe hypoxia occurred in 18% of adults tested and could not be predicted by examination or laboratory findings. Bacteremia was documented in 3.5% of episodes, but was strongly influenced by age (14% of infants and 1.8% of patients <10 years). ACS was most common in winter with children having the most striking increase. Transfusion was used less frequently, but earlier in children. Young children were hospitalized for 5.4 days versus 9 days for adults. Fifty percent of adults had a pain event in the 2 weeks preceding ACS and children were more likely to have febrile events. The death rate was four times higher in adults than in children. Fatal cases generally developed rapid pulmonary failure and one third were associated with bacteremia. Age has a striking effect on the clinical picture of ACS. In children, ACS was milder and more likely due to infection, whereas in adults ACS was severe, associated with pain and had a higher mortality rate.
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“Infection is the most common ACS trigger in children; common isolates include atypical microorganisms and Streptococcus pneumoniae (208).”
From Pathophysiology of Sickle Cell Disease (2019) · cites this paper as [208]
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“Elevated initial platelet count and dramatic drops during VOC have been found to portend a poor prognosis in ACS (29, 208, 219).”
From Pathophysiology of Sickle Cell Disease (2019) · cites this paper as [208]
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Armenis, I., Kalotychou, V., Tzanetea, R., Κollia, P., Kontogeorgiou, Z., Anastasopoulou, D., Mantzourani, M., Samarkos, M., Pantos, K., Konstantopoulos, K., et al. (2016) · Nitric Oxide · doi:10.1016/j.niox.2016.11.002 · Cited by 15
Leonard, A. K., Godiwala, N., Herrera, N. A., McCarter, R., Sharron, M. P., & Meier, E. R. (2018) · Blood Cells Molecules and Diseases · doi:10.1016/j.bcmd.2018.03.001 · Cited by 15
Transfusion practice for patients with sickle cell disease
Reed, W., & Vichinsky, E. P. (1999) · Current Opinion in Hematology · doi:10.1097/00062752-199911000-00014 · Cited by 15
Self-citationLonergan, G. J., Cline, D. B., & Abbondanzo, S. L. (2001) · Cited by 15
Transfusion Therapy: A Coming-of-Age Treatment for Patients With Sickle Cell Disease
Reed, W., & Vichinsky, E. P. (2001) · The American journal of pediatric hematology/oncology · doi:10.1097/00043426-200105000-00002 · Cited by 15
Self-citationMajor Changes in Sickle Cell Disease
Hagar, R. W., & Vichinsky, E. P. (2000) · Advances in Pediatrics · doi:10.1016/s0065-3101(23)00100-7 · Cited by 15
Self-citationBaseline and Disease-Induced Transcriptional Profiles in Children with Sickle Cell Disease
Creary, S. E., Shrestha, C. L., Kotha, K. S., Minta, A., Fitch, J., Jaramillo, L., Zhang, S., Pinto, S. J., Thompson, R., Ramilo, O., et al. (2020) · Scientific Reports · doi:10.1038/s41598-020-65822-3 · Cited by 14
Gbotosho, O. T., Gollamudi, J., & Hyacinth, H. I. (2023) · Biomolecules · doi:10.3390/biom13020381 · Cited by 15
“It is defined as the presence of fever and/or new respiratory symptoms such as cough, chest pain, and presence of a new pulmonary infiltrate on chest X-ray [16].”
BackgroundRespiratory management of acute chest syndrome in children with sickle cell disease
Ahmed, B., Arigliani, M., & Gupta, A. K. (2024) · European Respiratory Review · doi:10.1183/16000617.0005-2024 · Cited by 15
Bavle, A. A., Raj, A. B., Kong, M., & Bertolone, S. J. (2014) · Pediatric Blood & Cancer · doi:10.1002/pbc.25153 · Cited by 14
The relative resistance of children to sepsis mortality: from pathways to drug candidates
Joachim, R. B., Altschuler, G. M., Hutchinson, J. N., Wong, H. R., Hide, W. A., & Kobzik, L. (2018) · Molecular Systems Biology · doi:10.15252/msb.20177998 · Cited by 14
Lewing, K. B., Britton, K., DeBaun, M. R., & Woods, G. M. (2011) · Journal of Pediatric Hematology/Oncology · doi:10.1097/mph.0b013e31820994d0 · Cited by 14
The Acute Chest Syndrome of Sickle Cell Disease
(2000) · New England Journal of Medicine · doi:10.1056/nejm200008243430824 · Cited by 15
Madhi, F., Kamdem, A., Jung, C., Carlier‐Gonod, A., Biscardi, S., Busca, J., Arnaud, C., Hau, I., Narbey, D., Epaud, R., et al. (2019) · Journal of Clinical Medicine · doi:10.3390/jcm8111839 · Cited by 13
“First, the main studies identifying risk factors associated with increased incidence of ACS analyzed the data on ACS episodes in general, regardless of the etiology [3,4], whereas our study focused on ACS episodes that developed over the clinical course of a VOE.”
BackgroundGuenther, C. S., Pae, V. J., Neri, C. M., Barry, K., Duggan, M. A., & Cohen, R. T. (2021) · Pediatric Blood & Cancer · doi:10.1002/pbc.29136 · Cited by 13
Sickle Cell Disease Pathophysiology and Related Molecular and Biophysical Biomarkers
Saah, E. N., Fadaei, P., Gurkan, U. A., & Sheehan, V. (2022) · Hematology/Oncology Clinics of North America · doi:10.1016/j.hoc.2022.06.005 · Cited by 14
Acute anemic events in sickle cell disease
Reed, W., Walker, P., Haddix, T. L., & Perkins, H. A. (2000) · Transfusion · doi:10.1046/j.1537-2995.2000.40030267.x · Cited by 13
A Review of Acute Chest Syndrome in Pediatric Sickle Cell Disease
Abbas, H. A., Kahale, M., Hosn, M. A., & Inati, A. C. (2013) · Pediatric Annals · doi:10.3928/00904481-20130222-11 · Cited by 13
Clinical Manifestations of Sickle Cell Disease Across the Lifespan
Pecker, L. H., & Little, J. A. (2017) · Springer eBooks · doi:10.1007/978-3-319-62328-3_1 · Cited by 14
Badaki‐Makun, O., Casella, J. F., Tackett, S. A., Tao, X., & Chamberlain, J. M. (2020) · Annals of Emergency Medicine · doi:10.1016/j.annemergmed.2020.08.011 · Cited by 13
Hematopoietic cell transplantation for sickle cell disease: state of the art
Krishnamurti, L. (2007) · Expert Opinion on Biological Therapy · doi:10.1517/14712598.7.2.161 · Cited by 13
“Repeated events have been associated with an increased risk of chronic lung disease and early death [17].”
BackgroundAcute Chest Syndrome in Sickle Cell Disease in Saudi Arab Children in the Eastern Province
Al-Dabbous, I. A. (2002) · Annals of Saudi Medicine · doi:10.5144/0256-4947.2002.167 · Cited by 13
Crawford, M. W., Speakman, M., Carver, E. D., & Kim, P. C. W. (2004) · Canadian Journal of Anesthesia/Journal canadien d anesthésie · doi:10.1007/bf03018430 · Cited by 13
Metadata
- Authors
- Vichinsky, E. P., Styles, L. A., Colangelo, L. H., Wright, E. C., Castro, O., Nickerson, B., & Disease, T. C. S. O. S. C.
- Affiliations
- Children's Hospital of Orange County (US)
- Venue
- Blood
- Publisher
- American Society of Hematology
- Published
- 1997-03-01
- Volume
- 89
- Issue
- 5
- Pages
- 1787-1792
- ISSN
- 0006-4971 (ISSN-L)
- 1528-0020
- OpenAlex
- W1272191569
- Cited by
- 573
- References
- 30
- Citations by year
- 2026: 13 · 2025: 18 · 2024: 13 · 2023: 17 · 2022: 22 · 2021: 23 · 2020: 20 · 2019: 27 · 2018: 22 · 2017: 20 · 2016: 32 · 2015: 22 · 2014: 26 · 2013: 28 · 2012: 25
- FWCI
- 10.28
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Blood (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Genetics (clinical) › Hemoglobinopathies and Related Disorders
- Health Sciences › Medicine › Epidemiology › Pneumonia and Respiratory Infections
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Malaria Research and Control
- Keywords
- acute chest syndrome, sickle cell disease, clinical features, age differences, bacteremia, hypoxia, respiratory failure, pain, fever, hospital course, mortality, seasonal variation, blood transfusion
- MeSH terms
- Adolescent, Adult, Aged, Anemia, Sickle Cell, Chest Pain, Child, Child, Preschool, Humans, Infant, Middle Aged, Prospective Studies, Respiratory Insufficiency, Syndrome
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