Point-of-care ultrasound for the diagnosis of Swyer-James-MacLeod syndrome in pediatric emergency setting: a case report

Anna Maria Musolino; Lorenzo Di Sarno; Anya Caroselli; Nicola Ullmann; Paolo Maria Salvatore Schingo; Danilo Buonsenso; Antonio Chiaretti; Alberto Villani

doi:10.23750/abm.v95i6.15840

Point-of-care ultrasound for the diagnosis of Swyer-James-MacLeod syndrome in pediatric emergency setting: a case report

Authors

Anna Maria Musolino Pediatric Emergency Department, Bambino Gesù Children’s Hospital, Institute for Research and Health Care, IRCCS, Rome, Italy https://orcid.org/0000-0001-9606-2860
Lorenzo Di Sarno Department of Pediatrics, Fondazione Policlinico Universitario A. Gemelli, IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy https://orcid.org/0000-0002-4837-9297
Anya Caroselli Department of Pediatrics, Fondazione Policlinico Universitario A. Gemelli, IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy https://orcid.org/0009-0004-0254-1115
Nicola Ullmann Pediatric Pulmonology & Cystic Fibrosis Unit, Bambino Gesù Children’s Hospital, IRCCS, Rome, Italy https://orcid.org/0000-0003-1111-5690
Paolo Maria Salvatore Schingo Department of Imaging, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy https://orcid.org/0000-0003-3585-5677
Danilo Buonsenso Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy; Centro di Salute Globale, Università Cattolica del Sacro Cuore, Rome, Italy https://orcid.org/0000-0001-8567-2639
Antonio Chiaretti Department of Pediatrics, Fondazione Policlinico Universitario A. Gemelli, IRCCS, Rome, Italy https://orcid.org/0000-0002-9971-1640
Alberto Villani Pediatric Emergency Department, Bambino Gesù Children’s Hospital, Institute for Research and Health Care, IRCCS, Rome, Italy https://orcid.org/0000-0002-9120-0424

DOI: https://doi.org/10.23750/abm.v95i6.15840

Keywords:

POCUS, children, ultrasound, lung, emphysema

Abstract

Swyer–James–MacLeod syndrome is a rare emphysematous disease characterized by decreased pulmonary vascularity and hyperinflation that are secondary to repeated childhood respiratory infections. We present a case of a 2-years old boy admitted to our pediatric emergency ward for dyspnea and fever with clinical and radiological suspicion of bronchopneumonia. The patient was monitored by using lung point-of-care ultrasound. These lung evaluations showed an increased representation of pulmonary A-lines and absence of B-lines as from hyperinflation. Although the clinical conditions improved, this unconventional imaging, not compatible with pneumonia, triggered us to perform further examinations. Thus, a chest CT scan was executed showing a reduction in parenchymal density of the left lung and hence definitely diagnosing Swyer–James–MacLeod syndrome. This report shows how the smart and skilled use of lung point-of-care ultrasound is able to detect potential “red flags” that could suggest the execution of further examinations, avoiding diagnostic delays and thus changing the patient’s clinical history even in rare findings such as Swyer–James–MacLeod syndrome.

References

Omar M, Saeed MA, Patil A. Swyer-James-Macleod Syndrome: Case Report and Brief Literature Review. S D Med 2019; 72: 518-520. PMID: 31985903.

Chaucer B, Chevenon M, Toro C, Lemma T, Grageda M. Swyer-James-Macleod syndrome: a rare finding and important differential in the ED setting. Am J Emerg Med 2016; 34: 1329.e3-4. doi: 10.1016/j.ajem.2015.12.045.

Fregonese L, Girosi D, Battistini E et al. Clinical, physiologic, and roentgenographic changes after pneumonectomy in a boy with Macleod/Swyer-James syndrome and bronchiectasis. Pediatr Pulmonol 2002; 34: 412-416. doi: 10.1002/ppul.10178.

Behrendt A, Lee Y. Swyer-James-MacLeod Syndrome. Treasure Island (FL): StatPearls Publishing, 2023.

Swyer PR, James GC. A case of unilateral pulmonary emphysema. Thorax 1953; 8: 133-136. doi: 10.1136/thx.8.2.133.

Damle NA, Mishra R, Wadhwa JK. Classical imaging triad in a very young child with swyer-james syndrome. Nucl Med Mol Imaging 2012; 46: 115-118. doi: 10.1007/s13139-012-0131-2.

Dirweesh A, Alvarez C, Khan M, Shah N. A unilateral hyperlucent lung - Swyer-James syndrome: A case report and literature review. Respir Med Case Rep 2017; 20: 104-106. doi: 10.1016/j.rmcr.2017.01.004.

Moore AD, Godwin JD, Dietrich PA, Verschakelen JA, Henderson WR Jr. Swyer-James syndrome: CT findings in eight patients. AJR Am J Roentgenol 1992; 158: 1211-1215. doi: 10.2214/ajr.158.6.1590109.

Khalil KF, Saeed W. Swyer-James-MacLeod Syndrome. J Coll Physicians Surg Pak 2008; 18: 190-192. PMID: 18460255.

Altinsoy B, Altintas N. Diagnostic approach to unilateral hyperlucent lung. JRSM Short Rep 2011; 2: 95. doi: 10.1258/shorts.2011.011080.

Soldati G, Prediletto R, Demi M, Salvadori S, Pistolesi M. Operative Use of Thoracic Ultrasound in Respiratory Medicine: A Clinical Study. Diagnostics (Basel) 2022; 12: 952. doi: 10.3390/diagnostics12040952.

Aslan N, Yildizdas D, Horoz OO, Ekinci F, Study-Group TP. Point-of-care ultrasound use in pediatric intensive care units in Turkey. Turk J Pediatr 2020; 62: 770-777. doi: 10.24953/turkjped.2020.05.008.

Le Coz J, Orlandini S, Titomanlio L, Rinaldi VE. Point of care ultrasonography in the pediatric emergency department. Ital J Pediatr 2018; 44: 87. doi: 10.1186/s13052-018-0520-y.

Di Sarno L, Gatto A, Korn D et al. Pain management in pediatric age. An update. Acta Biomed 2023; 94: e2023174. doi: 10.23750/abm.v94i4.14289.

Singh Y, Tissot C, Fraga MV et al. International evidence-based guidelines on Point of Care Ultrasound (POCUS) for critically ill neonates and children issued by the POCUS Working Group of the European Society of Paediatric and Neonatal Intensive Care (ESPNIC). Crit Care 2020; 24: 65. doi: 10.1186/s13054-020-2787-9.

Finkel L, Ghelani S, Paladugu K, Sanyal S, Colla JS. General Pediatric Clinical Applications of POCUS: Part 2. Pediatr Ann 2020; 49: e196-e200. doi: 10.3928/19382359-20200319-02.

Brenner DJ, Hall EJ. Computed tomography--an increasing source of radiation exposure. N Engl J Med 2007; 357: 2277-2284. doi: 10.1056/NEJMra072149.

Ammirabile A, Buonsenso D, Di Mauro A. Lung Ultrasound in Pediatrics and Neonatology: An Update. Healthcare (Basel) 2021; 9: 1015. doi: 10.3390/healthcare9081015.

Rambhia SH, D'Agostino CA, Noor A, Villani R, Naidich JJ, Pellerito JS. Thoracic Ultrasound: Technique, Applications, and Interpretation. Curr Probl Diagn Radiol 2017; 46: 305-316. doi: 10.1067/j.cpradiol.2016.12.003.

Soldati G, Demi M, Smargiassi A, Inchingolo R, Demi L. The role of ultrasound lung artifacts in the diagnosis of respiratory diseases. Expert Rev Respir Med 2019; 13: 163-172. doi: 10.1080/17476348.2019.1565997.

Musolino AM, Di Sarno L, Buonsenso D et al. Use of POCUS for the assessment of dehydration in pediatric patients-a narrative review. Eur J Pediatr 2023; Epub ahead of print. doi: 10.1007/s00431-023-05394-2.

Musolino AM, Tomà P, De Rose C et al. Ten Years of Pediatric Lung Ultrasound: A Narrative Review. Front Physiol 2022; 12: 721951. doi: 10.3389/fphys.2021.721951.