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Ann Thorac Surg 2002;74:2226
© 2002 The Society of Thoracic Surgeons
a Section of Pediatric CardiologyChildren’s Hospital University of Colorado 1056 East 19th Avenue Denver CO 80218, USA
To the Editor:
Kaddoura and associates are to be commended for their innovative and aggressive approach to a daunting clinical problem supported with only minimal clinical experience and largely unknown outcomes [1].
We, Drs Todd (now deceased), Tinguely, and Norberg addressed the same difficult problem and published our intervention and its early results [2]. Here we provide additional details of treatment and report the survival of this index patient into adulthood. Our method utilized the methacrylate prosthesis in the midsternal position.
We clearly concluded that, when the skeletal limitation was eliminated, lung organ growth would occur. Late patient follow-up demonstrated that even though rib motility remains limited, the improvement in thoracic cage dimensions allows diapraghmatic motion to meet the respiratory demands of the patient.
Changes to larger prostheses to accommodate lung and patient growth were made without difficulty, but the need for a much better prosthesis and one that could be expanded was clearly evident. We believed that the early phase required extensive support of the sternal edges to prevent pressure necrosis but that later another pattern of prosthetic support would be required.
The last prosthesis developed a low-grade staphylococcal infection and was removed to treat the infection. A firm fibrous capsule had formed posterior to the prothesis and prevented cardiac and lung herniation. This fibrotic closure seemed adequate, and no further surgical interventions were done. Thoracic enlargement (growth) continued to meet patient respiratory and general growth requirements. I believe much of our difficulty related directly to the barotrauma and the resultant extremely severe bronchopulmonary dysplasia which was a concomitant problem of mechanical ventilation in that era. This patient received months of high pressure and high oxygen therapy before the first operation.
The late result is gratifying. The patient attended school, has been weaned off long-term ventilation, and is now able to pursue a somewhat physically limited but normal intellectual life compromised more by the phalangeal deformities of the condition than by the thoracic limitation. The severe scarring will require plastic surgical intervention to improve the cosmetic aspects.
I believe that aggressive innovative approaches by committed physicians and publication of procedural details combined with outcome information can lead to a solution to the problem of thoracic skeletal dystrophy causing pulmonary failure in Jeune’s and other forms of thoracic malformations.
References
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