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Correspondence

Case 30-1997: Pulmonary Interstitial Emphysema in Infancy

N Engl J Med 1998; 338:688-690March 5, 1998

Article

To the Editor:

In his discussion of Case 30-1997 (Sept. 25 issue),1 Dr. Wilson reviewed a number of treatments for diffuse pulmonary interstitial emphysema in infancy. Another option in cases of larger focal interstitial air collections is that of percutaneous evacuation under computed tomographic guidance. We used this approach in a case of pulmonary interstitial emphysema that developed in a full-term infant who received mechanical ventilation for pulmonary hypertension and persistent fetal circulation. A large amount of air collected interstitially in the right lung, and multiple cystic lesions appeared in the left upper lobe (Figure 1AFigure 1Large Pneumatocele at the Base of the Right Lung in a Full-Term Infant with Pulmonary Interstitial Emphysema Secondary to Mechanical Ventilation for Persistent Fetal Circulation (Panel A) and Evacuation of the Pneumatocele (Panel B).). When the largest of the collections in the right lung apparently began to interfere with mechanical ventilation, we elected to evacuate it percutaneously under computed tomographic guidance. This was accomplished with an 8-French pigtail catheter (Figure 1B). There was no recurrence when the catheter was removed after several days of suction. The child made an uneventful recovery.

Percutaneous evacuation of pneumatoceles and lung abscesses has been described in older children.2 This case demonstrates that this approach is also useful in selected neonatal patients.

Richard B. Fox, M.D.
Alan M. Wright, M.D.
Good Samaritan Hospital, San Jose, CA 95124

2 References
  1. 1

    Case Records of the Massachusetts General Hospital (Case 30-1997). N Engl J Med 1997;337:916-924
    Full Text | Web of Science | Medline

  2. 2

    Zuhdi MK, Spear RM, Worthen HM, Peterson BM. Percutaneous catheter drainage of tension pneumatocele, secondarily infected pneumatocele, and lung abscess in children. Crit Care Med 1996;24:330-333
    CrossRef | Web of Science | Medline

To the Editor:

The options listed by Wilson and Mark for treating pulmonary interstitial emphysema in newborns should have included dexamethasone. In 1987, Mohsini et al.1 reported three cases in which acquired lobar emphysema resolved after five to seven days of dexamethasone therapy at a dose of 0.5 mg per kilogram of body weight per day in respirator-dependent infants born at less than 30 weeks of gestation. The presumed mode of action of dexamethasone was an antiinflammatory effect, which is the rationale for its use in bronchopulmonary dysplasia, to relieve ball-valve obstruction within the airway. Another case2 of acquired giant lobar emphysema was cured by a three-day course of dexamethasone at a dose of 0.5 mg per kilogram per day in a triplet born at 31 weeks of gestation who was receiving mechanical ventilation. In our unit, my colleagues and I have used short courses of dexamethasone effectively to treat pulmonary interstitial emphysema (Figure 1Figure 1Results of Dexamethasone Therapy in a 790-g Twin Born at 25 Weeks of Gestation with Surfactant-Treated Hyaline Membrane Disease.) since the publication of the report by Mohsini et al.1 Two placebo-controlled, randomized trials showed early beneficial effects of dexamethasone given in the first days of life in infants with bronchopulmonary dysplasia.3,4 Before resorting to surgery for acquired lobar emphysema, physicians may find that a short course of dexamethasone is worth trying.

Francine Lefebvre, M.D.
Hôpital Sainte-Justine, Montreal, QC H3T 1C5, Canada

4 References
  1. 1

    Mohsini K, Reid D, Tanswell K. Resolution of acquired lobar emphysema with dexamethasone therapy. J Pediatr 1987;111:901-904
    CrossRef | Web of Science | Medline

  2. 2

    Saint-Martin J, Choulot JJ, Mensire A, Jasper E, Lugassy B, Hernandorena X. Emphyseme lobaire geant acquis chez un premature sous ventilation artificielle: guerison par la corticotherapie. Ann Pediatr (Paris) 1993;40:49-50
    Medline

  3. 3

    Rastogi A, Akintorin SM, Bez ML, Morales P, Pildes RS. A controlled trial of dexamethasone to prevent bronchopulmonary dysplasia in surfactant-treated infants. Pediatrics 1996;98:204-210
    Web of Science | Medline

  4. 4

    Sanders RJ, Cox C, Phelps DL, Sinkin RA. Two doses of early intravenous dexamethasone for the prevention of bronchopulmonary dysplasia in babies with respiratory distress syndrome. Pediatr Res 1994;36:122-128
    CrossRef | Web of Science | Medline

To the Editor:

Wilson and Mark discuss a preterm newborn with pulmonary interstitial emphysema. The crucial feature of their presentation was that pulmonary interstitial emphysema developed unilaterally in an infant who had not received mechanical ventilation but rather only nasal continuous positive airway pressure. Localized pulmonary interstitial emphysema has not previously been reported in association with nasal continuous positive airway pressure. Diagnostic and therapeutic procedures led to a right pneumonectomy. We describe a preterm newborn with a similar clinical presentation who fully recovered with a more conservative approach.

A 2290-g boy was born at 33 weeks of gestation. The Apgar scores were 6, 9, and 10 at 1, 5, and 10 minutes, respectively. Because of retractions and grunting 15 minutes after birth, nasal continuous positive airway pressure was instituted, and the infant was transferred to our intensive care unit.

A radiograph of the chest at two hours of age showed mild respiratory distress syndrome. The clinical status was stable, but retractions and tachypnea persisted. The oxygen need increased from 25 to 30 percent and a second chest radiograph obtained at 27 hours revealed interstitial emphysema of the right lung. One hour later, the infant's condition deteriorated and intubation and mechanical ventilation were instituted. A chest radiograph confirmed the presence of a pneumothorax on the right side, and a chest tube was placed. The course was complicated by repeated pneumothoraxes. The infant was treated with two doses of a natural surfactant and high-frequency oscillatory ventilation. Successful weaning and extubation were possible on day 10. Supplementary oxygen was necessary until day 26. Chest radiographs showed persistent cystic changes of the right lung, with a shift of the mediastinum to the left. Despite this complicated course, the boy fully recovered, with no abnormalities on the chest radiograph at the time of discharge (day 41).

Our case confirms that localized pulmonary interstitial emphysema may occur in preterm infants treated only by nasal continuous positive airway pressure. Since this method is increasingly used to avoid mechanical ventilation,1 clinicians should be aware of its complications. Our case also shows that a conservative approach in a condition that may be reversible can save an infant's lung. Expectant management instead of an aggressive approach may be warranted in preterm infants with cystic changes of the lung.

Claudia Roll, M.D.
Ludwig Hanssler, M.D.
Thomas Voit, M.D.
University Children's Hospital, 45122 Essen, Germany

1 References
  1. 1

    Verder H, Robertson B, Greisen G, et al. Surfactant therapy and nasal continuous positive airway pressure for newborns with respiratory distress syndrome. N Engl J Med 1994;331:1051-1055
    Full Text | Web of Science | Medline

Author/Editor Response

Dr. Wilson replies:

To the Editor: Fox and Wright offer another provocative option for the management of large focal interstitial emphysema. My colleagues and I have no experience with draining these collections percutaneously. However, as long as no clinically significant bronchopleural fistula ensues, I do not think that it is unreasonable to try the procedure in selected cases.

In response to Lefebvre's suggestion that a short course of dexamethasone may be useful in patients with pulmonary interstitial emphysema: at our institution, we do not use dexamethasone in the treatment of pulmonary interstitial emphysema, because we believe that this condition differs from chronic bronchopulmonary dysplasia. In patients with chronic lung disease who need increasing ventilatory support, we have used steroid treatment as a last resort.

With regard to the suggestion from Roll and colleagues that a conservative approach to pulmonary interstitial emphysema is preferable, I agree completely. In my discussion I tried to make it clear that we do treat pulmonary interstitial emphysema expectantly with surfactant therapy, high-frequency ventilation, and even extracorporeal membrane oxygenation to avoid the removal of the affected lung at all costs. In my retrospective review of the case, I concluded that pulmonary interstitial emphysema was the most likely diagnosis. However, having been told that a procedure was performed, I presumed that uncertainty existed as to the correct diagnosis. Consequently, I suggested a lung biopsy to distinguish between pulmonary interstitial emphysema and congenital cystic adenomatoid malformation, which I presumed was the other diagnosis being entertained. However, I agree that if localized pulmonary interstitial emphysema is the clear diagnosis, a nonoperative approach should be used and is usually successful.

Jay M. Wilson, M.D.
Children's Hospital, Boston, MA 02115

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