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Ann Thorac Surg 1996;61:1618-1624
© 1996 The Society of Thoracic Surgeons

Original Article: General Thoracic

Chest Wall Constriction After Too Extensive and Too Early Operations for Pectus Excavatum

J. Alex Haller, Jr, MD, Paul M. Colombani, MD, C. Thomas Humphries, MD, Richard G. Azizkhan, MD, Gerald M. Loughlin, MD

Divisions of Pediatric Surgery and Pediatric Pulmonary Medicine, The Johns Hopkins Children's Center, Baltimore, Maryland


   Abstract
Top
 Footnotes
 Abstract
Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
Background and Methods. Since 1990 we have evaluated 12 children and teenagers in whom severe cardiorespiratory symptoms have developed due to failure of chest wall growth after very extensive pectus excavatum operations (removal of five or more ribs) at very early ages (<4 years).

Results. Apparently these extensive procedures have removed or prevented growth center activity, which resulted in restriction of chest wall growth with marked limitation of ventilatory function. The forced vital capacity ranged from 30% to 50% of predicted and the forced expiratory volume in 1 second from 30% to 60%. All patients are symptomatic with mild exercise and cannot compete in running games. Our protocol for critical evaluation includes exercise pulmonary function studies and axial computed tomographic reconstruction.

Conclusions. This report is an alert to recognize such patients and also to recommend delay in operative repair in small children until at least 6 to 8 years of age. The younger the patient the more limited the chest wall resection for pectus excavatum should be. Five of these patients have had a chest cavity expansion operation with encouraging early results.


   Introduction
Top
 Footnotes
 Abstract
 Introduction
Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
See also page 1625.

In the past 5 years (1990 to 1995) we have evaluated 12 children and teenagers with severe symptoms of respiratory distress with exercise who had extensive pectus excavatum repair in early childhood and who have demonstrated failure of chest wall growth. All of these children had operative procedures with the removal of five or more rib cartilages on each side and all were operated on at very early ages (before 4 years of age). As older children and teenagers, all of these patients are symptomatic with mild exercise and cannot compete in running games and sports.

The purposes of this report are: (1) to alert pediatricians and surgeons to recognize patients with this complication of pectus repair, (2) to describe the symptomatology and describe the physical findings in this group of children and teenagers, (3) to recommend resting and exercise pulmonary function studies in the evaluation of these patients, (4) to offer a possible explanation for this failure of chest wall growth and development, (5) to describe an operative technique for relief of chest wall constriction by thoracic cavity expansion, and (6) to report early, preliminary postoperative studies that document partial relief of pulmonary functional abnormalities.


   Patient Group
Top
 Footnotes
 Abstract
 Introduction
 Patient Group
Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
As noted in Table 1Go, all 12 patients had been operated on for pectus excavatum deformities before 4 years of age; the majority (7) were 3 years of age or younger. All had very extensive rib cartilage excision as a part of their repair, usually the whole cartilage of five or six ribs on each side. Parents typically reported that their child's chest did not appear to enlarge after the operation with general body growth. The children became progressively short of breath with mild to moderate exercise and eventually they all sought advice because of growth retardation accentuated by this progressive decrease in exercise tolerance. None of the patients could compete in running games or sports. Several of the patients had repeated bouts of pneumonia, which were difficult to treat and had prolonged duration, suggesting some interference with normal pulmonary dynamics.


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  		Table 1. . Twelve Patients Who Were Operated on Before 4 Years of Age Who Presented With Severe Chest Wall Constriction and Symptoms of Pulmonary Dysfunction With Exercise
 

   Physical Findings
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 Abstract
 Introduction
 Patient Group
 Physical Findings
Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
The universal physical findings were a small, immovable anterior chest wall, a very long torso, primary diaphragmatic breathing, and no evidence of recurrence of the excavatum deformity (Fig 1Go). These findings immediately brought to mind the characteristic features of the prototype of chest wall growth retardation, namely, the congenital abnormality of asphyxiating thoracic chondrodystrophy or Jeune's syndrome [1–3]. This congenital pulmonary and chest wall growth abnormality usually causes death of the newborn infant because of respiratory insufficiency due to hypoplasia of the lungs and a very small chest. A few of these babies will survive with less severe forms of this congenital abnormality, and as shown in Figure 2Go, they may present with increasing respiratory distress and limitation of activity in early childhood. Most of these children ultimately succumb to repeated upper respiratory infections and are severely limited in their exercise tolerance. Often they become unsaturated with exercise.




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  		Fig 1. . This 12-year-old patient had pectus repair at age 2 years. He had no chest wall growth, and is unable to compete as a little league baseball pitcher because of shortness of breath. (Reprinted by permission of The Society of Thoracic Surgeons [Ann Thorac Surg 1995;60:1857–8].)

 



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  		Fig 2. . Six-year-old boy with congenital asphyxiating chondrodystrophy or Jeune's syndrome. He had marked limitation of exercise ability and desaturation while running.

 
Because the findings in this group of postoperative children are so similar to this congenital abnormality, we have labeled this condition of restrictive complications of extensive pectus excavatum repair "acquired Jeune's syndrome" (Figs 1, 3GoGo).




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  		Fig 3. . This 19-year-old patient had pectus repair at 3 years 3 months. He is unable to work because of shortness of breath climbing stairs. There is no anterior chest wall movement on respiration.

 

   Laboratory Tests
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 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
Representative chest roentgenograms and computed tomographic scans of some of these children are shown in Figure 4Go. We have also found that spiral computed tomographic reconstruction of the chest is helpful for three-dimensional evaluation of these children. These spatial scans are useful in estimating pulmonary volume and comparing postoperative results (Figure 5Go).




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  		Fig 4. . Computed tomograms of 2 patients with severe chest wall constriction (acquired Jeune's syndrome). Note rib regrowth behind sternum.

 



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  		Fig 5. . Examples of spatial computed tomographic reconstruction of patients with acquired Jeune's syndrome.

 
The most critical tests in evaluating these children are pulmonary function studies at rest and with maximal exercise [4–6]. The findings of these pulmonary function studies are shown in Table 2Go. Basically, these children and teenagers show a marked decrease in functional vital capacity and forced expiratory volume in 1 second. They also show marked decreases in exercise pulmonary function. In several children, desaturation occurred during these exercise pulmonary function studies.


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  		Table 2. . Pulmonary Function Studies in 12 Patients With Acquired Jeune's Syndromea
 

   Explanation for "Acquired Jeune's Syndrome"
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 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
It seems very likely that an extensive operative procedure at a very young age has significantly altered the growth potential of the chest wall. We would, therefore, propose the following explanation for growth retardation. These children were operated on at much younger ages (less than 4 years of age) than usually recommended (6 to 8 years) [7, 8]. At this young age, the various operative steps may have interfered more with the delicate growth centers of the chest wall, including the sternum, than would be true in later childhood and the early teenage years. Many more rib cartilages and much larger segments of these cartilages were removed in these children than is standard [7, 9]. Resection was often three to eight ribs on each side or five to six total rib cartilages bilaterally. The usual recommendation for the basic operative procedure is resection of 2.5-cm segments of three to four cartilages on each side, preserving the perichondrium [8].

In a subgroup of children who were operated on by a single surgeon, the rib cartilage and perichondrium of the second rib were removed and at that level a transverse osteotomy completely through the sternum bone was carried out. Possibly the growth center of the sternum was transected. In a standard operative procedure only the anterior cortex of the sternum is transected, not both, so this more extensive procedure may have interfered with the blood supply of the sternum and impeded growth and development.

Finally, in completing reconstruction of the lower chest wall, many of the children had the perichondral sheaths of rib seven, and often rib eight, sutured together bilaterally and brought beneath the sternum to give additional support. This substernal perichondral bridge was then sutured to the sternum. In so doing, when the cartilage regenerated and ultimately was replaced by bone, there would be marked restriction of the lower chest wall because of this semicirculer band of cartilage-bone (see Fig 4Go). This explanation is theoretical, because specific dynamics of growth of the anterior chest wall, including the growth centers for the sternum and costal cartilages, have not been well established.


   Proposed Operative Correction
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 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
Assuming that our understanding of this complication of very early extensive pectus excavatum repair was correct, we reasoned that it might be possible to improve pulmonary function, although we could not enhance pulmonary parenchymal growth at this late time in childhood and the teenage years. We developed an operative procedure that will expand the anterior chest wall and increase the volume of the chest cavity. In this way the trapped lung, which can only function by diaphragmatic excursion, might be freed for better diaphragmatic function. Characteristically, all of these children had very low-lying diaphragms.

As shown in Figures 6 through 11GoGoGoGoGoGo, we removed the regenerated cartilage/bone of the anterior ribs along the sternum, which allowed the sternum to be freed and brought anteriorally. Modified Rehbein splints (which have been used for decades to treat primary pectus excavatum in Europe) were modified and used as elevated bridges to hold the sternum in the new position and expand the anterior chest wall. The ends of the Rehbein splints were inserted in the marrow cavity of an appropriate pair of ribs and the two halves were brought together in front of the sternum. With various means of fixation to the sternum, this bow of stainless steel was constructed.



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  		Fig 6. . Figures 6 through 11GoGoGoGoGoGo show the proposed operative procedure to correct chest wall constriction in patients with acquired Jeune's syndrome.

 


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  		Fig 7. . Resection of abnormal costal cartilages. Prophylactic antibiotics are given. Sutured 7-8 rib perichondrium can be recognized behind the sternum.

 


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  		Fig 8. . Extensive mobilization of the sternum.

 


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  		Fig 9. . Insertion of Rehbein splints into appropriate bony ribs.

 


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  		Fig 10. . Construction of Rehbein splint bow with attachment to elevated sternum.

 


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  		Fig 11. . Realignment of perichondral sheaths to sternum, and placement of deep and subcutaneous drains.

 
In the initial operative procedure a transverse skin incision was used, but with extensive mobilization of the chest wall, it was impossible to bring together the anterior pectoral-rectus fascia without using prosthetic material as a gusset (Fig 12Go) Therefore, the incision was changed in the later children to a vertical incision because the pectoralis major muscle mass could be stretched and brought over the expanded sternum without the use of foreign material (Fig 13Go).



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  		Fig 12. . Gusset of woven silicone material to expand anterior chest wall fascia.

 





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  		Fig 13. . Second generation of proposed operative procedure for acquired Jeune's syndrome.

 
These children as a group have had remarkably smooth postoperative recoveries in spite of our concern about their basic lack of pulmonary functional reserve. We believe this rapid recovery may represent immediate improvement in pulmonary function, which is possibly best explained by the mechanical relief of diaphragmatic displacement, which permits better excursion.


   Postoperative Studies
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 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
Conclusions
 Addendum
 Acknowledgments
 References
 
Early postoperative studies in 6 patients show no change in resting functional vital capacity, which is not surprising if our theoretic explanation of the functional abnormality is correct, namely, interference with diaphragmatic function (see Table 2Go). All of the children are symptomatically improved and preliminary exercise pulmonary function studies show mild to moderate improvement 3 to 6 months after operation. It is far too soon to evaluate the total effect of chest wall expansion, but our early results are encouraging.


   Conclusions
Top
 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
Addendum
 Acknowledgments
 References
 
Extensive operative procedures for repair of pectus excavatum in very early infancy and childhood can interfere with chest wall growth and result in severe pulmonary dysfunction. This complication can be prevented by delaying repair until 6 to 8 years of age and by limiting the operative procedure to the resection of short segments of three or four cartilages on each side combined with a minimal sternal osteotomy.

We would strongly recommend that children with pectus excavatum deformities not be operated on in early infancy and childhood because there are no obvious advantages, and in rare instances, chest wall growth may be impaired [10].

We believe the mechanism of pulmonary functional improvement after operation in these children with acquired Jeune's syndrome is expansion of the thoracic cavity, which allows for better excursion and function of the diaphragm. The preliminary results of our reconstructive procedure suggest improved function. This operation may be the prototype for a corrective operative procedure for acquired Jeune's syndrome.


   Addendum
Top
 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
Acknowledgments
 References
 
Since this report was written (October 1995) 6 more (total, 11) children and teenagers with acquired Jeune's syndrome have had chest wall expansion operations. There were no immediate postoperative complications, but the Rehbein splints broke in 2 patients (at 5 months and 6 months) and required removal. In 2 patients the splint eroded through very thin chest skin with very little muscle coverage. Both required removal of the bar. In 1 patient the Rehbein splint pulled loose from the sternum and had to be reattached. Clearly, the operation is still in evolution.


   Acknowledgments
Top
 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
References
 
We thank Ms Karen Jelus for her careful measurements of exercise pulmonary function, which were performed with her characteristic sensitive professional techniques.


   Footnotes
Top
 Footnotes
Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 
Presented at the Forty-second Annual Meeting of the Southern Thoracic Surgical Association, San Antonio, TX, Nov 9-11, 1995.

Address reprint requests to Dr Haller, Division of Pediatric Surgery, Johns Hopkins Children's Center, 600 N Wolfe St, CMSC 7-113, Baltimore, MD 21287-3716.


   References
Top
 Footnotes
 Abstract
 Introduction
 Patient Group
 Physical Findings
 Laboratory Tests
 Explanation for "Acquired...
 Proposed Operative Correction
 Postoperative Studies
 Conclusions
 Addendum
 Acknowledgments
 References
 

  1. Jeune M, Carron R, Beraud C, et al. Polychondrodystrophie avec blocage thoracique d'volution fatale. Pediatric 1954;9:390–2.
  2. Kaufman HJ, Kirkpatrick JA. Jeune's thoracic dysplasia: a spectrum of disorders? In: Bergsma D, ed. Birth defects: original article series. Miami: Symposia Specialists, 1974;10(9):101–16.
  3. Pirnar R, Neuhauser EBD. Asphyxiating thoracic dystrophy of the newborn. AJR 1966;98:358–64.[Abstract/Free Full Text]
  4. Polgar G, Promadhat V, eds. Pulmonary function testing in children: techniques and standards. Philadelphia, Saunders: 1971.
  5. Wasserman K, Hansen JE, Sue DY, Wipp BJ. Measurements of the physiological response to exercise. In: Principles of exercise testing and interpretation. Philadelphia: Lea & Febiger, 1987:27–46.
  6. Cahill JL, Lees GM, Robertson HT. A summary of preoperative and postoperative cardiorespiratory performance in patients undergoing pectus excavatum and carinatum repair. J Pediatr Surg 1984;19:430–3.[Medline]
  7. Ravitch MM. Depression deformities. In: Welch KJ, Randolph JG, Ravitch MM, O'Neill JA, Rowe MI, eds. Pediatric surgery (4th ed). Chicago: Year Book Medical, 1986:568–78.
  8. Haller JA, Scherer LR, Turner CC, Colombani PM. Evolving management of pectus excavatum based on a single institutional experience of 664 patients. Ann Surg 1989;209:578–83.[Medline]
  9. Haller JA. Operative management of chest wall deformities in children: unique contributions of Southern thoracic surgeons. Ann Thorac Surg 1988;46:4–12.[Medline]
  10. Nathanson I. Chest wall abnormalities. In: Loughlin GM, Eigen H, eds. Respiratory disease in children: diagnosis and management. Baltimore: Williams and Wilkins, 1994:533–43.

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