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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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ResultInfluentialMekontso Dessap, A., Deux, J.-F., Habibi, A., Abidi, N., Godeau, B., Adnot, S., Brun-Buisson, C., Rahmouni, A., Galacteros, F., & Maitre, B. (2014) · Thorax · doi:10.1136/thoraxjnl-2013-203775 · Cited by 50
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BackgroundSelf-citationLow adjusted‐dose acenocoumarol therapy in sickle cell disease: A pilot study
Schnog, J. B., Kater, A. P., Gillavry, M. R. M., Duits, A. J., Lard, L. R., van der Dijs, F. P. L., Brandjes, D. P. M., Cate, H. T., van Eps, L. W. S., & Rojer, R. A. (2001) · American Journal of Hematology · doi:10.1002/ajh.1175 · Cited by 38
Bennewitz, M. F., Watkins, S. C., & Sundd, P. (2014) · IntraVital · doi:10.4161/intv.29748 · Cited by 37
How I manage red cell transfusions in patients with sickle cell disease
Rees, D. C., Robinson, S. E., & Howard, J. (2018) · British Journal of Haematology · doi:10.1111/bjh.15115 · Cited by 37
“Surgery in SCD is associated with increased complications, particularly an increased risk of acute pain or ACS.”
BackgroundInfluentialAlabdulaali, M. K. (2007) · Annals of Thoracic Medicine · doi:10.4103/1817-1737.36550 · Cited by 37
Sarode, R., Ballas, S. K., Garcia, A., Kim, H. C., King, K. E., Sachais, B. S., & Williams, L. A. (2016) · Journal of Clinical Apheresis · doi:10.1002/jca.21511 · Cited by 37
“In the ACS in SCD Study 76% of the subjects were 19 years of age or younger.(31)”
BackgroundInduced Sputum versus Bronchoalveolar Lavage during Acute Chest Syndrome in Sickle Cell Disease
Lechapt, E., Habibi, A., Bachir, D., Galactéros, F., Schaeffer, A., Desvaux, D., Brochard, L. J., HOUSSET, B., Godeau, B., & Maître, B. (2003) · American Journal of Respiratory and Critical Care Medicine · doi:10.1164/rccm.200302-174oc · Cited by 36
“Diagnosis of ACS was based on the presence of fever or chest pain, combined with new pulmonary infiltrates on chest X-ray ( 19 , 20).”
BackgroundBedside Lung Ultrasound During Acute Chest Syndrome in Sickle Cell Disease
Razazi, K., Deux, J., de Prost, N., Boissier, F., Cuquemelle, É., Galactéros, F., Rahmouni, A., Maître, B., Brun‐Buisson, C., & Dessap, A. M. (2016) · Medicine · doi:10.1097/md.0000000000002553 · Cited by 36
“We also excluded children and our findings may not be applicable to ACS during infancy, especially as the distribution of lung consolidations differs in adults and children with ACS, young children having more often isolated upper and middle lobe disease than adults and less often lower lobe disease.(37) Future studie…”
ResultPolymerized human Hb use in acute chest syndrome: a case report
Lanzkron, S., Moliterno, A. R., Norris, E. J., Gould, S. A., Segal, J. B., Nuermberger, E. L., & Ness, P. M. (2002) · Transfusion · doi:10.1046/j.1537-2995.2002.00184.x · Cited by 36
Acute pulmonary complications of sickle cell disease
Knight‐Madden, J., & Greenough, A. (2013) · Paediatric Respiratory Reviews · doi:10.1016/j.prrv.2013.10.005 · Cited by 35
Complications of PORT-A-CATH® in patients with sickle cell disease
Alkindi, S. S. A., Matwani, S., Al-Maawali, A., Maskari, B. A., & Pathare, A. V. (2011) · Journal of Infection and Public Health · doi:10.1016/j.jiph.2011.10.004 · Cited by 35
Reagan, M. M., DeBaun, M. R., & Frei‐Jones, M. J. (2010) · Pediatric Blood & Cancer · doi:10.1002/pbc.22808 · Cited by 35
HODGKIN'S DISEASE IN A CHILD WITH SICKLE CELL DISEASE TREATED WITH HYDROXYUREA
Moschovi, M. A., Psychou, F., Menegas, D., Tsangaris, G. T., Tzortzatou-Stathopoulou, F. F., & Nikolaidou, P. (2001) · Pediatric Hematology and Oncology · doi:10.1080/088800101316921985 · Cited by 35
Sickle cell disease in children: an update of the evidence in low- and middle-income settings
Odame, I. (2022) · Archives of Disease in Childhood · doi:10.1136/archdischild-2021-323633 · Cited by 36
“28 ACS can develop rapidly progressing from a mild- to-severe respiratory illness (with significant decline in haemoglobin and/or oxygen saturations) requiring ventilatory support.”
BackgroundMouse models of sickle cell disease: Imperfect and yet very informative
Kamimura, S., Smith, M. L., Vogel, S., Almeida, L. E. F., Thein, S. L., & Quezado, Z. (2023) · Blood Cells Molecules and Diseases · doi:10.1016/j.bcmd.2023.102776 · Cited by 37
Sickle Cell Disease in the Emergency Department
Lovett, P. B., Sule, H. P., & Lopez, B. L. (2014) · Emergency Medicine Clinics of North America · doi:10.1016/j.emc.2014.04.011 · Cited by 34
Risk factors and prediction of outcomes in children and adolescents who have sickle cell anemia
Quinn, C. T., & Miller, S. T. (2004) · Hematology/Oncology Clinics of North America · doi:10.1016/j.hoc.2004.07.004 · Cited by 34
The Role of RIB Infarcts in the Acute Chest Syndrome of Sickle Cell Diseases
Rucknagel, D. L. (2001) · Pediatric Pathology & Molecular Medicine · doi:10.1080/15513810109168607 · Cited by 32
Steady-state sVCAM-1 serum levels in adults with sickle cell disease
Schnog, J. B., Rojer, R. A., Gillavry, M. R. M., Cate, H. T., Brandjes, D. P. M., & Duits, A. J. (2003) · Annals of Hematology · doi:10.1007/s00277-003-0609-1 · Cited by 33
Nouraie, M. S., & Gordeuk, V. R. (2015) · Transfusion · doi:10.1111/trf.13155 · Cited by 32
Anesthesia and Hemoglobinopathies
Firth, P. G. (2009) · Anesthesiology Clinics · doi:10.1016/j.anclin.2009.05.001 · Cited by 33
Teixeira, R., Terse-Ramos, R., Ferreira, T. A., Machado, V. R., Perdiz, M. I., Lyra, I. M., Nascimento, V. M. L., Boa‐Sorte, N., Andrade, B. B., & Ladeia, A. M. T. (2017) · PLoS ONE · doi:10.1371/journal.pone.0184076 · Cited by 31
Hereditary Red Blood Cell Disorders in Middle Eastern Patients
Steensma, D. P., Hoyer, J. D., & Fairbanks, V. F. (2001) · Mayo Clinic Proceedings · doi:10.4065/76.3.285 · Cited by 31
Breathing Patterns During Vaso-occlusive Crisis of Sickle Cell Disease
Needleman, J. P., Benjamin, L. J., Sykes, J. A., & Aldrich, T. K. (2002) · CHEST Journal · doi:10.1378/chest.122.1.43 · Cited by 31
Johnson, C. S. (2005) · Hematology/Oncology Clinics of North America · doi:10.1016/j.hoc.2005.08.001 · Cited by 31
Willen, S. M., Rodeghier, M., ROSEN, C. L., & DeBaun, M. R. (2017) · American Journal of Hematology · doi:10.1002/ajh.25013 · Cited by 31
“58C, decreased SpO2 from baseline, or increased respiratory rate.(11,12) A new diagnosis of pneumonia was considered an ACS episode.”
BackgroundTakahashi, T., Okubo, Y., & Handa, A. (2017) · Pediatric Blood & Cancer · doi:10.1002/pbc.26885 · Cited by 30
Sickle Acute Lung Injury: Role of Prevention and Early Aggressive Intervention Strategies on Outcome
Melton, C. W., & Haynes, J. (2006) · Clinics in Chest Medicine · doi:10.1016/j.ccm.2006.04.001 · Cited by 30
Filho, I. L. D. S., Ribeiro, G. S., de Moura, P. G., Vechi, M. L., Cavalcante, A. C., & Andrada‐Serpa, M. J. (2012) · Revista Brasileira de Hematologia e Hemoterapia · doi:10.5581/1516-8484.20120049 · Cited by 30
Steen, R. G., Langston, J. W., Ogg, R. J., Xiong, X., Ye, Z., & Wang, W. C. (1999) · Magnetic Resonance Imaging · doi:10.1016/s0730-725x(98)00204-5 · Cited by 30
Ventilator-associated Pneumonia and the Gastropulmonary Route of Infection: A Pendulum
Bonten, M. J. M. (2011) · American Journal of Respiratory and Critical Care Medicine · doi:10.1164/rccm.201108-1537ed · Cited by 30
Daswani, D. D., Shah, V. P., Avner, J. R., Manwani, D. G., Kurian, J., & Rabiner, J. E. (2016) · Academic Emergency Medicine · doi:10.1111/acem.13002 · Cited by 29
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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