- Email: [email protected]
Left mainstem bronchopexy for severe bronchomalacia
Left Mainstem
Bronchopexy ByAnn
for Severe Bronchomalacia M. Kosloske
Albuquerque, New Mexico l Severe bronchomalacia occurred in a lCmonth-old boy, as a result of compression of the left mainstem bronchus by a bronchogenic cyst. After resection of the cyst, the bronchomalacia was corrected by suspension of the posterolateral bronchial wall to the ligamentum arteriosum. This method of bronchopexy may be of value for severe left mainstem bronchomalacia. Copyright o 1991 by W.B. Saunders Company INDEX WORDS:
Bronchomalacia;
bronchopexy.
C
OLLAPSE OF A major airway in a child may be surgically correctable. Although aortopexy is usually curative for severe tracheomalacia,‘22 no standard operation exists for bronchomalacia. This report describes an infant with severe left mainstem bronchomalacia secondary to compression from a bronchogenic cyst, who was successfully treated by a bronchopexy procedure. CASE REPORT A 14-month-old boy was transferred to the Children’s Hospital of New Mexico with left-lung atelectasis. He had had respiratory distress and left-lung emphysema at birth. Bronchomalacia was diagnosed at bronchoscopy. The respiratory distress resolved, and he was discharged at 21 days of age. Nasal oxygen was subsequently discontinued, and he thrived at home. However, at 12 months of age he was treated at a local hospital for respiratory distress and cough. Left lower lobe atelectasis, attributed to reactive airway disease, responded to treatment with bronchodilators. Respiratory distress recurred a month later, and chest x-ray showed atelectasis of the entire left lung (Fig 1). The lung did not reexpand after 3 days of intensive treatment with physiotherapy, bronchodilators, antibiotics, and steroids. Physical findings on admission to the Children’s Hospital of New Mexico included absent breath sounds and dullness to percussion over the entire left hemithorax. At bronchoscopy, the left mainstem bronchus was completely occluded (Fig 2A), but the Storz 3.5mm bronchoscope could be gently advanced through the collapsed area into a normalappearing bronchus distally. The left upper lobe orifice, lower lobe bronchus, and segmental bronchi were normal. The segment of collapse was about 1.5 cm long, extending from the carina to just above the take-off of the left upper lobe bronchus. Computed tomography (CT) scan showed a large cystic mediastinal mass. At thoracotomy, a 4-cm bronchogenic cyst was resected from beneath
From the Division of Pediatric Surgery, Department of Surgery, Universiry of New Mexico School of Medicine, Albuquerque, NM. Presented at the 2Ist Annual Meeting of the American Pediatric Surgical Association, Vancouver, British Columbia, May 19-22, 1990. Address reprint requests to Ann M. Kosloske, MD, 36 Talbott Ave, Timonium, MD 21093. Copyright o 1991 by WB. Saunders Company 0022-3468/91/2603-0004$03.00/0
260
Fig 1. Preoperative chest x-ray showing atelectasis with herniation of right lung across the midline.
of left lung,
the arch of the aorta. The left mainstem bronchus was flattened on its posterolateral aspect by compression from the deep portion of the cyst. Positive-pressure ventilation reexpanded the atelectatic left lung, but as closure of the chest began, the anesthesiologist noted decreased compliance, and the left lung became emphysematous and rigid. These changes were attributed to severe acute bronchomalacia. Therefore, bronchopexy was performed, with suspension of the posterolateral wall of the left mainstem bronchus to the ligamentum arteriosum. After bronchopexy, compliance of the left lung was much improved; it filled and emptied with ventilation. Repeat bronchoscopy following closure of the chest showed a patent left mainstem bronchus (Fig 2B). Postoperative chest x-ray was normal (Fig 3). He made an uneventful recovery and is thriving and healthy 16 months after operation. A chest x-ray at 14 months after operation showed mild left-sided emphysema. The operative technique is depicted in Fig 4.
Fig 2. Bronchoscopic views showing left mainstem bronchus: (A) collapsed before bronchopexy, and (B) patent after bronchopexy. (Original magnification x6.1
Journal of Pediatric Surgery, Vol26, No 3 (March), 1991:
pp 260-262
MAINSTEM
BRONCHOPEXY
Fig 3.
261
FOR BRONCHOMAIACIA
Postoperative
Fig 4. Operative procedure viewed at left posterolateral thoracotomy, depicting (A) bronchogenic cyst with compression of left mainstem bronchus (inset), and (B) technique of left mainstem bronchopexy. Four monofilament (Prolene) sutures were placed, two directly lateral, and two posterolateral, to elevate bronchus (inset). Care was taken to protect the recurrent laryngeal nerve.
normal chest x-ray.
DISCUSSION
The first report of a corrective operation for the collapsing pediatric airway, by Gross and Newhauser in 1948, described aortopexy for treatment of tracheomalacia from innominate artery compression.3 The softened cartilage of tracheomalacia is most often associated with congenital esophageal atresia and tracheoesophageal fistula,4-6 a vascular ring, or a mediastinal mass. Surgical treatment is indicated only in severely symptomatic cases. Aortopexy, which elevates the anterior wall of the trachea, is usually successful. Several other alternatives include variants of tracheopexy,’ external airway splinting,‘.8,9 wedge tracheoplasty,” or tracheostomy for internal splinting of the collapsing airway. The surgical alternatives for bronchomalacia are limited. Reports from the 1960s concluded that pneumonectomy or lobectomy was necessary because segmental bronchial resection with end-to-end anastomosis was not feasible in a young child.“,” Filler et al, in
1982, reported successful splinting of a left mainstem bronchus in a 3%year-old girl with congenital bronchomalacia, using a splint of Marlex mesh supported by Silastic ribs and rings.* The ligamenturn arteriosum is a fibrous cord that connects the left pulmonary artery to the arch of the aorta. It is formed by obliteration of the lumen of the ductus arteriosus, usually during the first week of life. The anatomical proximity of the ductus arteriosus to the left mainstem bronchus may, in rare instances, be a disadvantage; a severe rightward mediastinal shift may produce occlusion of the bronchus by the ductus following right pneumonectomy’3 or with congenital agenesis of the right lung.14 However, in this patient the proximity of the two structures was advantageous. The ligamenturn was fibrous, strong enough to hold the suspension, and in a perfect location for bronchopexy. This procedure may be of value in patients with severe bronchomalacia of the left mainstem bronchus.
REFERENCES 1. Blair GK, Cohen R, Filler RM: Treatment of tracheomalacia: Eight years’ experience. J Pediatr Surg 21:781-7851986 2. Greenholz SK, Karrer FM, Lilly JR: Contemporary surgery of tracheomalacia. J Pediatr Surg 21:511-514,1986 3. Gross RE, Newhauser EBD: Compression of the trachea by an anomalous innominate artery-An operation for its relief. Am J Dis Child 75:570-574, 1948 4. Benjamin B, Cohen D, Glasson M: Tracheomalacia in association with congenital tracheoesophageal fistula. Surgery 79:504508,1976 5. Filler RM, Rossello PJ, Lebowitz RL: Life-threatening anoxic spells caused by tracheal compression after repair of esophageal atresia. J Pediatr Surg 11:739-748, 1976
6. Wailoo MP, Emery JL: The trachea in children with tracheooesophageal fistula. Histopathology 3:329-338, 1979 7. Conti VR, Lobe TE: Vascular sling with tracheomalacia: Surgical management. Ann Thorac Surg 47:310-311,1989 8. Filler RM, Buck JR, Bahoric A, et al: Treatment of segmental tracheomalacia and bronchomalacia by implantation of an airway splint. J Pediatr Surg 17:597-603, 1982 9. Vinograd I, Filler RM, Bahoric A: Long-term functional results of prosthetic airway splinting in tracheomalacia and bronchomalacia. J Pediatr Surg 22:38-41, 1987 10. Hernandez-Cano AM, Lilly JR: Wedge tracheoplasty infants. Surg Gynecol Obstet 165:277-278. 1987
in
262
ANN M. KOSLOSKE
11. Gupta TGCM, Goldberg SJ, Lewis E, et al: Congenital bronchomalacia. Am J Dis Child 115:88-90.1968 12. MacMahon HE, Ruggieri J: Congenital chomalacia. Am J Dis Child 118:923-926, 1969
segmental bron-
13. Stolar C, Berdon W, Reyes C, et al: Right pneumonectomy
syndrome: A lethal complication of lung resection in a newborn with cvstic adenomatoid malformation. J Pediatr Sum 23:11801183, i988 14. Harrison MR, Hendren WH: Agenesis of the lung complicated by vascular compression and bronchomafacia. J Pediatr Surg 10:813-817,1975
Discussion R Filler (Toronto, Ontario): We performed bronchopexy using a different procedure in a child with severe left mainstem bronchomalacia. Instead of suspending the bronchus superiorly to the ligamenturn arteriosum, as Dr Kosloske has done, we suspended it posteriorly to the adventitia of the descending aorta. The procedure worked well. Each case of bronchomalacia is a little different, and I think one needs to be guided by the anatomy. A.M. Kosloske (response): I appreciate Dr Filler’s comments, since it is he and his colleagues in Toronto
who have literally written the textbook on tracheoplastic and bronchoplastic procedures. The bronchoperq may not last forever, but it may last long enough to allow the airways to grow to a size at which bronchomalacia no longer produces severe symptoms. The suspension procedure certainly got us out of a difficult situation in which none of the other surgical options-sleeve resection, placement of a rigid prosthesis between aorta and pulmonary artery, or even pneumonectomy-seemed good. With the bronchopexy, we burned no bridges, and did no harm.
Bronchopexy ByAnn
for Severe Bronchomalacia M. Kosloske
Albuquerque, New Mexico l Severe bronchomalacia occurred in a lCmonth-old boy, as a result of compression of the left mainstem bronchus by a bronchogenic cyst. After resection of the cyst, the bronchomalacia was corrected by suspension of the posterolateral bronchial wall to the ligamentum arteriosum. This method of bronchopexy may be of value for severe left mainstem bronchomalacia. Copyright o 1991 by W.B. Saunders Company INDEX WORDS:
Bronchomalacia;
bronchopexy.
C
OLLAPSE OF A major airway in a child may be surgically correctable. Although aortopexy is usually curative for severe tracheomalacia,‘22 no standard operation exists for bronchomalacia. This report describes an infant with severe left mainstem bronchomalacia secondary to compression from a bronchogenic cyst, who was successfully treated by a bronchopexy procedure. CASE REPORT A 14-month-old boy was transferred to the Children’s Hospital of New Mexico with left-lung atelectasis. He had had respiratory distress and left-lung emphysema at birth. Bronchomalacia was diagnosed at bronchoscopy. The respiratory distress resolved, and he was discharged at 21 days of age. Nasal oxygen was subsequently discontinued, and he thrived at home. However, at 12 months of age he was treated at a local hospital for respiratory distress and cough. Left lower lobe atelectasis, attributed to reactive airway disease, responded to treatment with bronchodilators. Respiratory distress recurred a month later, and chest x-ray showed atelectasis of the entire left lung (Fig 1). The lung did not reexpand after 3 days of intensive treatment with physiotherapy, bronchodilators, antibiotics, and steroids. Physical findings on admission to the Children’s Hospital of New Mexico included absent breath sounds and dullness to percussion over the entire left hemithorax. At bronchoscopy, the left mainstem bronchus was completely occluded (Fig 2A), but the Storz 3.5mm bronchoscope could be gently advanced through the collapsed area into a normalappearing bronchus distally. The left upper lobe orifice, lower lobe bronchus, and segmental bronchi were normal. The segment of collapse was about 1.5 cm long, extending from the carina to just above the take-off of the left upper lobe bronchus. Computed tomography (CT) scan showed a large cystic mediastinal mass. At thoracotomy, a 4-cm bronchogenic cyst was resected from beneath
From the Division of Pediatric Surgery, Department of Surgery, Universiry of New Mexico School of Medicine, Albuquerque, NM. Presented at the 2Ist Annual Meeting of the American Pediatric Surgical Association, Vancouver, British Columbia, May 19-22, 1990. Address reprint requests to Ann M. Kosloske, MD, 36 Talbott Ave, Timonium, MD 21093. Copyright o 1991 by WB. Saunders Company 0022-3468/91/2603-0004$03.00/0
260
Fig 1. Preoperative chest x-ray showing atelectasis with herniation of right lung across the midline.
of left lung,
the arch of the aorta. The left mainstem bronchus was flattened on its posterolateral aspect by compression from the deep portion of the cyst. Positive-pressure ventilation reexpanded the atelectatic left lung, but as closure of the chest began, the anesthesiologist noted decreased compliance, and the left lung became emphysematous and rigid. These changes were attributed to severe acute bronchomalacia. Therefore, bronchopexy was performed, with suspension of the posterolateral wall of the left mainstem bronchus to the ligamentum arteriosum. After bronchopexy, compliance of the left lung was much improved; it filled and emptied with ventilation. Repeat bronchoscopy following closure of the chest showed a patent left mainstem bronchus (Fig 2B). Postoperative chest x-ray was normal (Fig 3). He made an uneventful recovery and is thriving and healthy 16 months after operation. A chest x-ray at 14 months after operation showed mild left-sided emphysema. The operative technique is depicted in Fig 4.
Fig 2. Bronchoscopic views showing left mainstem bronchus: (A) collapsed before bronchopexy, and (B) patent after bronchopexy. (Original magnification x6.1
Journal of Pediatric Surgery, Vol26, No 3 (March), 1991:
pp 260-262
MAINSTEM
BRONCHOPEXY
Fig 3.
261
FOR BRONCHOMAIACIA
Postoperative
Fig 4. Operative procedure viewed at left posterolateral thoracotomy, depicting (A) bronchogenic cyst with compression of left mainstem bronchus (inset), and (B) technique of left mainstem bronchopexy. Four monofilament (Prolene) sutures were placed, two directly lateral, and two posterolateral, to elevate bronchus (inset). Care was taken to protect the recurrent laryngeal nerve.
normal chest x-ray.
DISCUSSION
The first report of a corrective operation for the collapsing pediatric airway, by Gross and Newhauser in 1948, described aortopexy for treatment of tracheomalacia from innominate artery compression.3 The softened cartilage of tracheomalacia is most often associated with congenital esophageal atresia and tracheoesophageal fistula,4-6 a vascular ring, or a mediastinal mass. Surgical treatment is indicated only in severely symptomatic cases. Aortopexy, which elevates the anterior wall of the trachea, is usually successful. Several other alternatives include variants of tracheopexy,’ external airway splinting,‘.8,9 wedge tracheoplasty,” or tracheostomy for internal splinting of the collapsing airway. The surgical alternatives for bronchomalacia are limited. Reports from the 1960s concluded that pneumonectomy or lobectomy was necessary because segmental bronchial resection with end-to-end anastomosis was not feasible in a young child.“,” Filler et al, in
1982, reported successful splinting of a left mainstem bronchus in a 3%year-old girl with congenital bronchomalacia, using a splint of Marlex mesh supported by Silastic ribs and rings.* The ligamenturn arteriosum is a fibrous cord that connects the left pulmonary artery to the arch of the aorta. It is formed by obliteration of the lumen of the ductus arteriosus, usually during the first week of life. The anatomical proximity of the ductus arteriosus to the left mainstem bronchus may, in rare instances, be a disadvantage; a severe rightward mediastinal shift may produce occlusion of the bronchus by the ductus following right pneumonectomy’3 or with congenital agenesis of the right lung.14 However, in this patient the proximity of the two structures was advantageous. The ligamenturn was fibrous, strong enough to hold the suspension, and in a perfect location for bronchopexy. This procedure may be of value in patients with severe bronchomalacia of the left mainstem bronchus.
REFERENCES 1. Blair GK, Cohen R, Filler RM: Treatment of tracheomalacia: Eight years’ experience. J Pediatr Surg 21:781-7851986 2. Greenholz SK, Karrer FM, Lilly JR: Contemporary surgery of tracheomalacia. J Pediatr Surg 21:511-514,1986 3. Gross RE, Newhauser EBD: Compression of the trachea by an anomalous innominate artery-An operation for its relief. Am J Dis Child 75:570-574, 1948 4. Benjamin B, Cohen D, Glasson M: Tracheomalacia in association with congenital tracheoesophageal fistula. Surgery 79:504508,1976 5. Filler RM, Rossello PJ, Lebowitz RL: Life-threatening anoxic spells caused by tracheal compression after repair of esophageal atresia. J Pediatr Surg 11:739-748, 1976
6. Wailoo MP, Emery JL: The trachea in children with tracheooesophageal fistula. Histopathology 3:329-338, 1979 7. Conti VR, Lobe TE: Vascular sling with tracheomalacia: Surgical management. Ann Thorac Surg 47:310-311,1989 8. Filler RM, Buck JR, Bahoric A, et al: Treatment of segmental tracheomalacia and bronchomalacia by implantation of an airway splint. J Pediatr Surg 17:597-603, 1982 9. Vinograd I, Filler RM, Bahoric A: Long-term functional results of prosthetic airway splinting in tracheomalacia and bronchomalacia. J Pediatr Surg 22:38-41, 1987 10. Hernandez-Cano AM, Lilly JR: Wedge tracheoplasty infants. Surg Gynecol Obstet 165:277-278. 1987
in
262
ANN M. KOSLOSKE
11. Gupta TGCM, Goldberg SJ, Lewis E, et al: Congenital bronchomalacia. Am J Dis Child 115:88-90.1968 12. MacMahon HE, Ruggieri J: Congenital chomalacia. Am J Dis Child 118:923-926, 1969
segmental bron-
13. Stolar C, Berdon W, Reyes C, et al: Right pneumonectomy
syndrome: A lethal complication of lung resection in a newborn with cvstic adenomatoid malformation. J Pediatr Sum 23:11801183, i988 14. Harrison MR, Hendren WH: Agenesis of the lung complicated by vascular compression and bronchomafacia. J Pediatr Surg 10:813-817,1975
Discussion R Filler (Toronto, Ontario): We performed bronchopexy using a different procedure in a child with severe left mainstem bronchomalacia. Instead of suspending the bronchus superiorly to the ligamenturn arteriosum, as Dr Kosloske has done, we suspended it posteriorly to the adventitia of the descending aorta. The procedure worked well. Each case of bronchomalacia is a little different, and I think one needs to be guided by the anatomy. A.M. Kosloske (response): I appreciate Dr Filler’s comments, since it is he and his colleagues in Toronto
who have literally written the textbook on tracheoplastic and bronchoplastic procedures. The bronchoperq may not last forever, but it may last long enough to allow the airways to grow to a size at which bronchomalacia no longer produces severe symptoms. The suspension procedure certainly got us out of a difficult situation in which none of the other surgical options-sleeve resection, placement of a rigid prosthesis between aorta and pulmonary artery, or even pneumonectomy-seemed good. With the bronchopexy, we burned no bridges, and did no harm.