Pediatric Pulmonology 49:E90–E95 (2014)
Case Report On the Nature of Pleural Involvement in Necrotizing Pneumonia: A Report of Two Cases of Life Threatening Late Complications Nadir Demirel, MD,1* Annabelle Quizon, MD,1 Edgar Leonel Belteto n De Leon, MD,2 Joel Reiter, MD,1 1 and Andrew A. Colin, MD Summary. Suppurative complications of pneumonia such as empyema, lung abscess, pyopneumothorax, and necrotizing pneumonia (NP) are uncommon in children. Over the last decade an increasing incidence of NP has been reported. Streptococcus pneumoniae continues to be the predominant causative organism of NP, and while sporadic cases were reported prior to routine administration of heptavalent pneumococcal vaccine, a marked increase in NP appears to relate to replacement pneumococcal strains. Pleural involvement is almost universal in NP, and the course of pleural disease often determines its duration and outcome, particularly as it relates to complication of bronchopleural fistula. Cavities are formed in NP within the lung parenchyma and in the pleural space as the fibrosing pleural process organizes. The similarity of the radiologic appearance of parenchymal and pleural space cavities often makes the differentiation of pneumatocele versus residua of loculated pneumothorax challenging. The prevailing perception from most reports on childhood NP is of a favorable outcome with conservative approach. We report two pediatric cases with pneumonia who presented with prolonged fever despite antibiotic treatment, eventually diagnosed with NP. After stabilization on prolonged IV antibiotics, and weeks after discharge, they presented with unexpected acute respiratory failure due to a life-threatening tension air collection. In this article we discuss the nature of NP, its typical presentation, benign course and outcome, albeit its potential to cause serious late complications in the light of our recent experiences. Increasing awareness of such complications will result in more careful followup and in providing appropriate recommendations to parents of patients recovering from NP. Pediatr Pulmonol. 2014; 49:E90–E95. ß 2013 Wiley Periodicals, Inc. Key words: necrotizing pneumonia; pneumatocele; pneumothorax; bronchopleural fistula; complications. Funding source: none reported 1 Division of Pediatric Pulmonology, Department of Pediatrics, Miller School of Medicine, University of Miami, Miami, Florida. 2 Pediatric, Intensive Care, Pediatric Medicine, Centro Pediatrico de Guatemala, Guatemala, Guatemala.
Conflict of interest: None. Author contribution: Nadir Demirel, MD: collected clinical information, wrote the manuscript and responsible for final approval and submission of the manuscript. Annabelle Quizon, MD: conceptualized this report, critically reviewed and revised the manuscript, and approved the final manuscript as submitted. Edgar Leonel Belteto´n De Leon, MD: provided clinical information, reviewed the manuscript, and approved the final manuscript as submitted. Joel Reiter, MD: reviewed the manuscript and approved the final manuscript as submitted. Andrew A. Colin, MD: conceptualized this report, critically reviewed and revised the manuscript, and approved the final manuscript as submitted. What’s Known in This Subject? • An increasing incidence of complications of community-acquired pneumonia such as necrotizing pneumonia (NP) has been reported.
ß 2013 Wiley Periodicals, Inc.
Streptococcus pneumonia is the leading etiology despite introduction of pneumococcal vaccine. The prevailing perception is a favorable outcome of NP with conservative management. What the Report Adds? • NP may be complicated by development of pneumatoceles and loculated pneumothorax which can result in tension air collections (tension pneumothorax or tension pneumatocele) weeks after discharge. We report 2 cases of NP with life threatening late complication; provide putative mechanisms and recommendation for follow up.
Correspondence to: Nadir Demirel, MD, Division of Pediatric Pulmonology, Batchelor Children’s Research Institute, 1580 NW 10th Avenue, Miami, FL 33136. E-mail: [email protected] Received 26 April 2013; Accepted 3 September 2013. DOI 10.1002/ppul.22943 Published online 23 November 2013 in Wiley Online Library (wileyonlinelibrary.com).
Late Complications of Necrotizing Pneumonia
INTRODUCTION
Over the last decade an increasing incidence of necrotizing pneumonia (NP), also termed cavitatory pneumonia,1–3 has been reported as a complication of community-acquired pneumonia (CAP) in children.4–6 Streptococcus pneumoniae (SP) continues to be the predominant organism, and while sporadic cases were reported prior to routine administration of heptavalent pneumococcal vaccine (PCV7),4,6 a marked increase in NP appears to relate to replacement strains.5 This trend tracks an overall increase in incidence of complicated pneumonia attributed to this replacement phenomenon.7,8 Pleural involvement is almost universal in NP, and often determines duration and outcome of NP, particularly as it relates to complication of bronchopleural fistula (BPF).6,9 The prevailing perception on childhood NP is of a favorable outcome with conservative approach.4,6 We report an unusual late complication of NP in two pediatric patients who presented weeks after discharge with life-threatening air collections—tension pneumothorax (PT) or tension pneumatocele (PC). Our objective is to discuss the nature of the cavitary disease and its potential to cause serious late complications. Awareness of such complications will result in providing appropriate recommendations to parents of patients recovering from NP. CASE 1
A previously healthy 12-month-old female infant presented to the emergency room (ER) with a 5-dayhistory of cough and fever. Chest radiograph (CXR) revealed right (Rt) lower lobe opacity. She was discharged on amoxicillin with diagnosis of pneumonia. She returned 4 days later with dyspnea and persistent fever. CXR revealed pleural effusion and multiple lucencies with surrounding airspace disease in the Rt lung. Chest ultrasonogram revealed complex Rt-sided pleural effusion with septations and consolidation in the Rt lower lung and a small left-sided pleural effusion. She was admitted to the pediatric intensive care unit (PICU) with
ABBREVIATIONS: BPF Bronchopleural fistula CAP Community-acquired pneumonia CT Computerized tomography CXR Chest radiograph ER Emergency room NP Necrotizing pneumonia PC Pneumatocele PCV7 Heptavalent pneumococcal vaccine PICU Pediatric intensive care unit PT Pneumothorax Rt Right SP Streptococcus pneumonia Oxygen saturation SpO2
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the diagnosis of NP with empyema and received intravenous cefotaxime and vancomycin. She had an unremarkable birth and past medical history. Immunizations were up-to-date. Family history was non-contributory; no sick contacts. Laboratory tests showed WBC of 37,300/mm3 with left shift, CRP was increased at >27 mg/dl. Blood and respiratory cultures were negative. She defervesced and maintained normal oxygen saturation (SpO2) by Day-2, thus interventions were deferred. On Day-4, CXR showed multiple enlarging cavities but improvement of consolidations and pleural effusion. On Day-9 she developed respiratory distress with increase in the size of cavities on CXR. Chest computerized tomography (CT) showed multiple large cavities involving all lobes of the Rt lung with one fluid-filled cavity (Fig. 1a and b). Respiratory distress resolved spontaneously and laboratory markers continued to improve. She was discharged on Day-17 to complete intravenous antibiotics for 3 weeks. Four weeks after discharge, she developed fever, cough and coryza and brought to the ER in respiratory distress and with decreased breath sounds in the Rt lung. CXR showed left shift of the mediastinum with air collection in the Rt hemithorax (tension PT or PC) (Fig. 2). She was readmitted to the PICU and a pigtail catheter was inserted and attached to suction resulting in reexpansion of the Rt lung. She developed an air leak assumed to be secondary to BPF. She received antibiotics and defervesced by Day-3; blood culture was negative. Respiratory status improved and CXRs showed stable findings; the chest catheter was removed on Day-10. Repeat chest CT showed a small PT (Fig. 3). She was discharged to complete a 14-day course of antibiotics and remained stable on follow-up. CASE 2
An 18-month-old male infant with no significant past medical history developed cough and fever of 1-week duration treated with amoxicillin-clavulanate. He presented to the ER with fever, respiratory distress, and hypoxia with SpO2 of 88% on room air and decreased breath sounds in the Rt lung. CXR showed Rt paracardiac infiltrates and a large ipsilateral effusion. He was admitted to the PICU with a diagnosis of parapneumonic effusion and received IV cefotaxime and vancomycin. Birth history was unremarkable. Immunizations were up to date. Family history was non-contributory; no sick contacts. Laboratory tests showed WBC of 22,000/mm3 with left shift. Chest CT showed multi-loculated Rt pleural effusion and NP was considered; a chest tube was placed. Pleural fluid culture was positive for SP type 2. Chest tube was removed on day-6. Follow-up chest CT showed multiple pneumatoceles (PC) in the Rt (Fig. 4a and b). Pediatric Pulmonology
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Fig. 1. a, b: Chest CT shows multiple large cavities in the right lung with a fluid-filled cavity as shown in lung and mediastinal windows, respectively.
He was discharged after 10 days of intravenous antibiotics with recommendation for additional 2 weeks of oral cefuroxime. One week after discharge, he developed acute respiratory distress and fever. CXR showed left shift of the mediastinum with air collection in the Rt hemithorax (tension PT or PC) (Fig. 5a and b). He was readmitted to the PICU. A chest tube was inserted and attached to suction. By Day-3 there was no air leak and chest tube was removed; he was subsequently discharged. CXR 3 weeks later showed complete resolution of PT. DISCUSSION
Suppurative complications of pneumonia such as empyema, lung abscess, pyopneumothorax, and NP are uncommon in children. Over the last decade the rate of
Fig. 2. Right hemithorax air collection (pneumothorax/pneumatocele) with left shift of mediastinum.
Pediatric Pulmonology
complicated pneumonia has increased with resurgence of SP as the leading organism.5,7,10–15 A national study of hospitalization trends for pneumonia after universal introduction of PCV7 in 2000 showed a significant decrease in hospitalization for CAP for children below age 1, but a significant increase in older children; there was an increase in the incidence of local complications for all age groups.15 The increased incidence of NP was also reported by other investigators.1,5,6,16 While not fully settled8 the inference is that invasive disease is caused by replacement pneumococcal strains and the emergence of MRSA.17 Specifically in case 2, the SP was identified as type-2 not included in PCV7. The use of chest CT scans in characterizing complicated pneumonias has improved diagnosis and in fact, enabled the definition of NP.4 The question of whether the increased incidence of NP is attributable to increased use of CT through the 1990s6 was addressed by a recent report supporting a veritable increase in incidence.5 Since the initial publication by Kerem et al.4 larger pediatric case
Fig. 3. Chest CT before discharge showing residual small pneumothorax.
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Fig. 4. a and b: Chest CT showing multiple pneumatoceles in the right lung as shown in lung and mediastinal windows, respectively.
series provided better understanding of the natural history of NP.6,18 NP is typically indistinguishable from lobar pneumonia clinically and by CXR and ultimately the distinction between these two parenchymal processes is established when a CT scan is obtained. The acquisition of a CT is mostly triggered by a clinical scenario in which the patient remains symptomatic despite appropriate antibiotic treatment and drainage of the frequently associated pleural effusion.2 Less commonly diagnosis of NP is established during thoracoscopy for empyema.19 Importantly, the course and complications of pleural involvement frequently determine the length of hospitalization.6,20 Amongst these complications is BPF with persistent air leak that has been reported in 12% of cases of NP6 and as high as 66% in cases that underwent thoracoscopy.19 Hoffer et al.9 speculated that BPF results from friability of the visceral pleura abutting the necrotic lung with tendency to rupture with manipulations.
Cavities are formed in NP within the lung parenchyma and in the pleural space as the fibrosing pleural process organizes. The similarity of the appearance on CXR and even on chest CT scan of parenchymal and pleural space cavities often makes the differentiation of PC versus loculated PT challenging.21,22 An enlarging PC, particularly subpleural, may be easily confused with a loculated PT23 while pleural cavitations may be erroneously termed PC. The management of PC, and for that matter loculated PT, is almost universally conservative. Such cavities are widely viewed as self-resolving, and none of the studies in children in recent years advocate drainage,2,4,6,24 except when complicated by tension PC.25 Similarly, Joseph et al.26 recommend conservative management for PCs of all sizes. An exception is the recommendation for early drainage by Imamoglu et al.21 for tension PC and persistent or large PC occupying more than 50% of the hemithorax.
Fig. 5. a, b: Right hemithorax air collection (pneumothorax/pneumatocele) with left shift of the mediastinum as seen in frontal and lateral views, respectively.
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Tension PC or tension PT have been observed as early complications of NP appearing in the first few days of presentation.27,28 The assumed underlying mechanisms for the two processes are respectively that in tension PC a ballvalve physiology leads to air trapping and increased intracavitary pressure resulting in distention and impingement on adjacent areas gradually causing cardiorespiratory compromise.29 The likely mechanism underlying late presentation of tension PT in this scenario is that BPF persists after removal of the chest drain and results in a low-grade leak that slowly builds-up to tension PT. The clinical presentation of these separate pathologies may be indistinguishable. Both scenarios, however, appear to be uncommon and our review of the literature failed to reveal how frequently PCs (or NPs) complicate and how often this occurs late in the course. The two studies that addressed the problem mentioned this almost as an afterthought. In a study of 80 cases of NP, three developed small pneumothoraces post-discharge not requiring intervention.6 In a smaller study of 33 cases of PCs, one developed tension PT after discharge; details were not provided.28 SUMMARY AND CONCLUSIONS
We report two cases of NP who developed lifethreatening air collections (tension PT or PC) weeks after discharge and drove us to review the assumption of a universally benign outcome of patients recovering from NP. We speculate that slowly evolving accumulation of air in the pleural space from a residual BPF progresses to PT. Alternatively a ball valve mechanism may lead to increase in the size of intrapulmonary cavities resulting in tension PC. Airway infections may increase the risk of air accumulation with associated cough resulting in high intrathoracic pressure that exacerbates the underlying leak. We propose that children with residual cavities, either PCs or loculated PTs, ought to be followed with a CXR about 2 weeks after discharge. Recommendations should include warning to parents about a low risk for tension PT and tension PC. Parents should be instructed to detect worsening condition by observation of breathing pattern or count of respiratory rate and to seek medical attention if unexpected changes occur. REFERENCES 1. Ramphul N, Eastham KM, Freeman R, Eltringham G, Kearns AM, Leeming JP, Hasan A, Hamilton LJR, Spencer DA. Cavitatory lung disease complicating empyema in children. Pediatr Pulmonol 2006;41:750–753. 2. Fretzayas A, Moustaki M, Alexopoulou E, Nychtari G, Nicolaidou P, Priftis KN. Clinical notations on bacteremic cavitating pneumococcal pneumonia in nonvaccinated immunocompetent children. J Trop Pediatr 2009;55:257–261. 3. Yangco BG, Deresinski SC. Necrotizing or cavitating pneumonia due to Streptococcus pneumoniae: report of four cases and review of the literature. Medicine (Baltimore) 1980;59:449–457.
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4. Kerem E, Bar Ziv Y, Rudenski B, Katz S, Kleid D, Branski D. Bacteremic necrotizing pneumococcal pneumonia in children. Am J Respir Crit Care Med 1994;149:242–244. 5. Bender JM, Ampofo K, Korgenski K, Daly J, Pavia AT, Mason EO, Byington CL. Pneumococcal necrotizing pneumonia in Utah: does serotype matter? Clin Infect Dis 2008;46:1346–1352. 6. Sawicki GS, Lu FL, Valim C, Cleveland RH, Colin AA. Necrotising pneumonia is an increasingly detected complication of pneumonia in children. Eur Respir J 2008;31:1285–1291. 7. Tan TQ, Mason EO, Jr., Wald ER, Barson WJ, Schutze GE, Bradley JS, Givner LB, Yogev R, Kim KS, Kaplan SL. Clinical characteristics of children with complicated pneumonia caused by Streptococcus pneumoniae. Pediatrics 2002;110:1–6. 8. Weinberger DM, Malley R, Lipsitch M. Serotype replacement in disease after pneumococcal vaccination. Lancet 2011;378: 1962–1973. 9. Hoffer FA, Bloom DA, Colin AA, Fishman SJ. Lung abscess versus necrotizing pneumonia: implications for interventional therapy. Pediatr Radiol 1999;29:87–91. 10. Hodina M, Hanquinet S, Cotting J, Schnyder P, Gudinchet F. Imaging of cavitary necrosis in complicated childhood pneumonia. Eur Radiol 2002;12:391–396. 11. Wexler ID, Knoll S, Picard E, Villa Y, Shoseyov D, Engelhard D, Kerem E. Clinical characteristics and outcome of complicated pneumococcal pneumonia in a pediatric population. Pediatr Pulmonol 2006;41:726–734. 12. Hausdorff WP. Invasive pneumococcal disease in children: geographic and temporal variations in incidence and serotype distribution. Eur J Pediatr 2002;161:S135–139. 13. Hsieh YC, Hsueh PR, Lu CY, Lee PI, Lee CY, Huang LM. Clinical manifestations and molecular epidemiology of necrotizing pneumonia and empyema caused by Streptococcus pneumoniae in children in Taiwan. Clin Infect Dis 2004;38:830–835. 14. Byington CL, Spencer LY, Johnson TA, Pavia AT, Allen D, Mason EO, Kaplan S, Carroll KC, Daly JA, Christenson JC, Samore MH. An epidemiological investigation of a sustained high rate of pediatric parapneumonic empyema: risk factors and microbiological associations. Clin Infect Dis 2002;34:434–440. 15. Lee GE, Lorch SA, Sheffler-Collins S, Kronman MP, Shah SS. National hospitalization trends for pediatric pneumonia and associated complications. Pediatrics 2010;126:204–213. 16. Rees JH, Spencer DA, Parikh D, Weller P. Increase in incidence of childhood empyema in West Midlands, UK. Lancet 1997; 349:402. 17. Schultz KD, Fan LL, Pinsky J, Ochoa L, Smith EO, Kaplan SL, Brandt ML. The changing face of pleural empyemas in children: epidemiology and management. Pediatrics 2004;113:1735–1740. 18. Donnelly LF, Klosterman LA. The yield of CT of children who have complicated pneumonia and noncontributory chest radiography. AJR Am J Roentgenol 1998;170:1627–1631. 19. Macedo M, Meyer KF, Oliveira TC. Necrotizing pneumonia in children submitted to thoracoscopy due to pleural empyema: incidence, treatment and clinical evolution. J Bras Pneumol 2010; 36:301–305. 20. McCarthy VP, Patamasucon P, Gaines T, Lucas MA. Necrotizing pneumococcal pneumonia in childhood. Pediatr Pulmonol 1999; 28:217–221. 21. Imamoglu M, Cay A, Kosucu P, Ozdemir O, Cobanoglu U, Orhan F, Akyol A, Sarihan H. Pneumatoceles in postpneumonic empyema: an algorithmic approach. J Pediatr Surg 2005;40: 1111–1117. 22. Caffey J. On the natural regression of pulmonary cysts during early infancy. Pediatrics 1953;11:48–63. 23. Boisset GF. Subpleural emphysema complicating staphylococcal and other pneumonias. J Pediatr 1972;81:259–266.
Late Complications of Necrotizing Pneumonia 24. Murphy D, Lockhart CH, Todd JK. Pneumococcal empyema: outcome of medical management. Am J Dis Child 1980;134: 659–662. 25. Sewall LE, Franco AI, Wojtowycz MM, McDermott JC. Pneumatoceles causing respiratory compromise. Treatment by percutaneous decompression. Chest 1993;103:1266–1267. 26. Joseph L, Shahroor S, Fisher D, Goldberg S, Picard E. Conservative treatment of a large post-infectious pneumatocele. Pediatr Int 2010;52:841–843.
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27. Amitai I, Mogle P, Godfrey S, Aviad I. Pneumatocele in infants and children. Report of 12 cases. Clin Pediatr (Phila) 1983;22: 420–422. 28. Kunyoshi V, Cataneo DC, Cataneo AJ. Complicated pneumonias with empyema and/or pneumatocele in children. Pediatr Surg Int 2006;22:186–190. 29. Al-Saleh S, Grasemann H, Cox P. Necrotizing pneumonia complicated by early and late pneumatoceles. Can Respir J 2008;15:129–132.
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Case Report On the Nature of Pleural Involvement in Necrotizing Pneumonia: A Report of Two Cases of Life Threatening Late Complications Nadir Demirel, MD,1* Annabelle Quizon, MD,1 Edgar Leonel Belteto n De Leon, MD,2 Joel Reiter, MD,1 1 and Andrew A. Colin, MD Summary. Suppurative complications of pneumonia such as empyema, lung abscess, pyopneumothorax, and necrotizing pneumonia (NP) are uncommon in children. Over the last decade an increasing incidence of NP has been reported. Streptococcus pneumoniae continues to be the predominant causative organism of NP, and while sporadic cases were reported prior to routine administration of heptavalent pneumococcal vaccine, a marked increase in NP appears to relate to replacement pneumococcal strains. Pleural involvement is almost universal in NP, and the course of pleural disease often determines its duration and outcome, particularly as it relates to complication of bronchopleural fistula. Cavities are formed in NP within the lung parenchyma and in the pleural space as the fibrosing pleural process organizes. The similarity of the radiologic appearance of parenchymal and pleural space cavities often makes the differentiation of pneumatocele versus residua of loculated pneumothorax challenging. The prevailing perception from most reports on childhood NP is of a favorable outcome with conservative approach. We report two pediatric cases with pneumonia who presented with prolonged fever despite antibiotic treatment, eventually diagnosed with NP. After stabilization on prolonged IV antibiotics, and weeks after discharge, they presented with unexpected acute respiratory failure due to a life-threatening tension air collection. In this article we discuss the nature of NP, its typical presentation, benign course and outcome, albeit its potential to cause serious late complications in the light of our recent experiences. Increasing awareness of such complications will result in more careful followup and in providing appropriate recommendations to parents of patients recovering from NP. Pediatr Pulmonol. 2014; 49:E90–E95. ß 2013 Wiley Periodicals, Inc. Key words: necrotizing pneumonia; pneumatocele; pneumothorax; bronchopleural fistula; complications. Funding source: none reported 1 Division of Pediatric Pulmonology, Department of Pediatrics, Miller School of Medicine, University of Miami, Miami, Florida. 2 Pediatric, Intensive Care, Pediatric Medicine, Centro Pediatrico de Guatemala, Guatemala, Guatemala.
Conflict of interest: None. Author contribution: Nadir Demirel, MD: collected clinical information, wrote the manuscript and responsible for final approval and submission of the manuscript. Annabelle Quizon, MD: conceptualized this report, critically reviewed and revised the manuscript, and approved the final manuscript as submitted. Edgar Leonel Belteto´n De Leon, MD: provided clinical information, reviewed the manuscript, and approved the final manuscript as submitted. Joel Reiter, MD: reviewed the manuscript and approved the final manuscript as submitted. Andrew A. Colin, MD: conceptualized this report, critically reviewed and revised the manuscript, and approved the final manuscript as submitted. What’s Known in This Subject? • An increasing incidence of complications of community-acquired pneumonia such as necrotizing pneumonia (NP) has been reported.
ß 2013 Wiley Periodicals, Inc.
Streptococcus pneumonia is the leading etiology despite introduction of pneumococcal vaccine. The prevailing perception is a favorable outcome of NP with conservative management. What the Report Adds? • NP may be complicated by development of pneumatoceles and loculated pneumothorax which can result in tension air collections (tension pneumothorax or tension pneumatocele) weeks after discharge. We report 2 cases of NP with life threatening late complication; provide putative mechanisms and recommendation for follow up.
Correspondence to: Nadir Demirel, MD, Division of Pediatric Pulmonology, Batchelor Children’s Research Institute, 1580 NW 10th Avenue, Miami, FL 33136. E-mail: [email protected] Received 26 April 2013; Accepted 3 September 2013. DOI 10.1002/ppul.22943 Published online 23 November 2013 in Wiley Online Library (wileyonlinelibrary.com).
Late Complications of Necrotizing Pneumonia
INTRODUCTION
Over the last decade an increasing incidence of necrotizing pneumonia (NP), also termed cavitatory pneumonia,1–3 has been reported as a complication of community-acquired pneumonia (CAP) in children.4–6 Streptococcus pneumoniae (SP) continues to be the predominant organism, and while sporadic cases were reported prior to routine administration of heptavalent pneumococcal vaccine (PCV7),4,6 a marked increase in NP appears to relate to replacement strains.5 This trend tracks an overall increase in incidence of complicated pneumonia attributed to this replacement phenomenon.7,8 Pleural involvement is almost universal in NP, and often determines duration and outcome of NP, particularly as it relates to complication of bronchopleural fistula (BPF).6,9 The prevailing perception on childhood NP is of a favorable outcome with conservative approach.4,6 We report an unusual late complication of NP in two pediatric patients who presented weeks after discharge with life-threatening air collections—tension pneumothorax (PT) or tension pneumatocele (PC). Our objective is to discuss the nature of the cavitary disease and its potential to cause serious late complications. Awareness of such complications will result in providing appropriate recommendations to parents of patients recovering from NP. CASE 1
A previously healthy 12-month-old female infant presented to the emergency room (ER) with a 5-dayhistory of cough and fever. Chest radiograph (CXR) revealed right (Rt) lower lobe opacity. She was discharged on amoxicillin with diagnosis of pneumonia. She returned 4 days later with dyspnea and persistent fever. CXR revealed pleural effusion and multiple lucencies with surrounding airspace disease in the Rt lung. Chest ultrasonogram revealed complex Rt-sided pleural effusion with septations and consolidation in the Rt lower lung and a small left-sided pleural effusion. She was admitted to the pediatric intensive care unit (PICU) with
ABBREVIATIONS: BPF Bronchopleural fistula CAP Community-acquired pneumonia CT Computerized tomography CXR Chest radiograph ER Emergency room NP Necrotizing pneumonia PC Pneumatocele PCV7 Heptavalent pneumococcal vaccine PICU Pediatric intensive care unit PT Pneumothorax Rt Right SP Streptococcus pneumonia Oxygen saturation SpO2
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the diagnosis of NP with empyema and received intravenous cefotaxime and vancomycin. She had an unremarkable birth and past medical history. Immunizations were up-to-date. Family history was non-contributory; no sick contacts. Laboratory tests showed WBC of 37,300/mm3 with left shift, CRP was increased at >27 mg/dl. Blood and respiratory cultures were negative. She defervesced and maintained normal oxygen saturation (SpO2) by Day-2, thus interventions were deferred. On Day-4, CXR showed multiple enlarging cavities but improvement of consolidations and pleural effusion. On Day-9 she developed respiratory distress with increase in the size of cavities on CXR. Chest computerized tomography (CT) showed multiple large cavities involving all lobes of the Rt lung with one fluid-filled cavity (Fig. 1a and b). Respiratory distress resolved spontaneously and laboratory markers continued to improve. She was discharged on Day-17 to complete intravenous antibiotics for 3 weeks. Four weeks after discharge, she developed fever, cough and coryza and brought to the ER in respiratory distress and with decreased breath sounds in the Rt lung. CXR showed left shift of the mediastinum with air collection in the Rt hemithorax (tension PT or PC) (Fig. 2). She was readmitted to the PICU and a pigtail catheter was inserted and attached to suction resulting in reexpansion of the Rt lung. She developed an air leak assumed to be secondary to BPF. She received antibiotics and defervesced by Day-3; blood culture was negative. Respiratory status improved and CXRs showed stable findings; the chest catheter was removed on Day-10. Repeat chest CT showed a small PT (Fig. 3). She was discharged to complete a 14-day course of antibiotics and remained stable on follow-up. CASE 2
An 18-month-old male infant with no significant past medical history developed cough and fever of 1-week duration treated with amoxicillin-clavulanate. He presented to the ER with fever, respiratory distress, and hypoxia with SpO2 of 88% on room air and decreased breath sounds in the Rt lung. CXR showed Rt paracardiac infiltrates and a large ipsilateral effusion. He was admitted to the PICU with a diagnosis of parapneumonic effusion and received IV cefotaxime and vancomycin. Birth history was unremarkable. Immunizations were up to date. Family history was non-contributory; no sick contacts. Laboratory tests showed WBC of 22,000/mm3 with left shift. Chest CT showed multi-loculated Rt pleural effusion and NP was considered; a chest tube was placed. Pleural fluid culture was positive for SP type 2. Chest tube was removed on day-6. Follow-up chest CT showed multiple pneumatoceles (PC) in the Rt (Fig. 4a and b). Pediatric Pulmonology
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Fig. 1. a, b: Chest CT shows multiple large cavities in the right lung with a fluid-filled cavity as shown in lung and mediastinal windows, respectively.
He was discharged after 10 days of intravenous antibiotics with recommendation for additional 2 weeks of oral cefuroxime. One week after discharge, he developed acute respiratory distress and fever. CXR showed left shift of the mediastinum with air collection in the Rt hemithorax (tension PT or PC) (Fig. 5a and b). He was readmitted to the PICU. A chest tube was inserted and attached to suction. By Day-3 there was no air leak and chest tube was removed; he was subsequently discharged. CXR 3 weeks later showed complete resolution of PT. DISCUSSION
Suppurative complications of pneumonia such as empyema, lung abscess, pyopneumothorax, and NP are uncommon in children. Over the last decade the rate of
Fig. 2. Right hemithorax air collection (pneumothorax/pneumatocele) with left shift of mediastinum.
Pediatric Pulmonology
complicated pneumonia has increased with resurgence of SP as the leading organism.5,7,10–15 A national study of hospitalization trends for pneumonia after universal introduction of PCV7 in 2000 showed a significant decrease in hospitalization for CAP for children below age 1, but a significant increase in older children; there was an increase in the incidence of local complications for all age groups.15 The increased incidence of NP was also reported by other investigators.1,5,6,16 While not fully settled8 the inference is that invasive disease is caused by replacement pneumococcal strains and the emergence of MRSA.17 Specifically in case 2, the SP was identified as type-2 not included in PCV7. The use of chest CT scans in characterizing complicated pneumonias has improved diagnosis and in fact, enabled the definition of NP.4 The question of whether the increased incidence of NP is attributable to increased use of CT through the 1990s6 was addressed by a recent report supporting a veritable increase in incidence.5 Since the initial publication by Kerem et al.4 larger pediatric case
Fig. 3. Chest CT before discharge showing residual small pneumothorax.
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Fig. 4. a and b: Chest CT showing multiple pneumatoceles in the right lung as shown in lung and mediastinal windows, respectively.
series provided better understanding of the natural history of NP.6,18 NP is typically indistinguishable from lobar pneumonia clinically and by CXR and ultimately the distinction between these two parenchymal processes is established when a CT scan is obtained. The acquisition of a CT is mostly triggered by a clinical scenario in which the patient remains symptomatic despite appropriate antibiotic treatment and drainage of the frequently associated pleural effusion.2 Less commonly diagnosis of NP is established during thoracoscopy for empyema.19 Importantly, the course and complications of pleural involvement frequently determine the length of hospitalization.6,20 Amongst these complications is BPF with persistent air leak that has been reported in 12% of cases of NP6 and as high as 66% in cases that underwent thoracoscopy.19 Hoffer et al.9 speculated that BPF results from friability of the visceral pleura abutting the necrotic lung with tendency to rupture with manipulations.
Cavities are formed in NP within the lung parenchyma and in the pleural space as the fibrosing pleural process organizes. The similarity of the appearance on CXR and even on chest CT scan of parenchymal and pleural space cavities often makes the differentiation of PC versus loculated PT challenging.21,22 An enlarging PC, particularly subpleural, may be easily confused with a loculated PT23 while pleural cavitations may be erroneously termed PC. The management of PC, and for that matter loculated PT, is almost universally conservative. Such cavities are widely viewed as self-resolving, and none of the studies in children in recent years advocate drainage,2,4,6,24 except when complicated by tension PC.25 Similarly, Joseph et al.26 recommend conservative management for PCs of all sizes. An exception is the recommendation for early drainage by Imamoglu et al.21 for tension PC and persistent or large PC occupying more than 50% of the hemithorax.
Fig. 5. a, b: Right hemithorax air collection (pneumothorax/pneumatocele) with left shift of the mediastinum as seen in frontal and lateral views, respectively.
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Tension PC or tension PT have been observed as early complications of NP appearing in the first few days of presentation.27,28 The assumed underlying mechanisms for the two processes are respectively that in tension PC a ballvalve physiology leads to air trapping and increased intracavitary pressure resulting in distention and impingement on adjacent areas gradually causing cardiorespiratory compromise.29 The likely mechanism underlying late presentation of tension PT in this scenario is that BPF persists after removal of the chest drain and results in a low-grade leak that slowly builds-up to tension PT. The clinical presentation of these separate pathologies may be indistinguishable. Both scenarios, however, appear to be uncommon and our review of the literature failed to reveal how frequently PCs (or NPs) complicate and how often this occurs late in the course. The two studies that addressed the problem mentioned this almost as an afterthought. In a study of 80 cases of NP, three developed small pneumothoraces post-discharge not requiring intervention.6 In a smaller study of 33 cases of PCs, one developed tension PT after discharge; details were not provided.28 SUMMARY AND CONCLUSIONS
We report two cases of NP who developed lifethreatening air collections (tension PT or PC) weeks after discharge and drove us to review the assumption of a universally benign outcome of patients recovering from NP. We speculate that slowly evolving accumulation of air in the pleural space from a residual BPF progresses to PT. Alternatively a ball valve mechanism may lead to increase in the size of intrapulmonary cavities resulting in tension PC. Airway infections may increase the risk of air accumulation with associated cough resulting in high intrathoracic pressure that exacerbates the underlying leak. We propose that children with residual cavities, either PCs or loculated PTs, ought to be followed with a CXR about 2 weeks after discharge. Recommendations should include warning to parents about a low risk for tension PT and tension PC. Parents should be instructed to detect worsening condition by observation of breathing pattern or count of respiratory rate and to seek medical attention if unexpected changes occur. REFERENCES 1. Ramphul N, Eastham KM, Freeman R, Eltringham G, Kearns AM, Leeming JP, Hasan A, Hamilton LJR, Spencer DA. Cavitatory lung disease complicating empyema in children. Pediatr Pulmonol 2006;41:750–753. 2. Fretzayas A, Moustaki M, Alexopoulou E, Nychtari G, Nicolaidou P, Priftis KN. Clinical notations on bacteremic cavitating pneumococcal pneumonia in nonvaccinated immunocompetent children. J Trop Pediatr 2009;55:257–261. 3. Yangco BG, Deresinski SC. Necrotizing or cavitating pneumonia due to Streptococcus pneumoniae: report of four cases and review of the literature. Medicine (Baltimore) 1980;59:449–457.
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