Pediatric Endosurgery & Innovative Techniques Volume 6, Number 1, 2002 © Mary Ann Liebert, Inc.
Case Report Pectus Carinatum as a Sequela of Minimally Invasive Pectus Excavatum Repair ANDRÉ HEBRA, M.D., PATRICK B. THOMAS, M.D., EDWARD P. TAGGE, M.D., WILLIAM T. ADAMSON, M.D., and H. BIEMANN OTHERSEN, M.D.
ABSTRACT Reported complications of minimally invasive repair of pectus excavatum (MIRPE) include bar displacement, pneumothorax, pleural effusion, and, rarely, thoracic outlet syndrome, pericarditis, major blood loss, cardiac dysrhythmias, cardiac injury, sternal erosion, and pseudoaneurysm. A 13-year-old boy with severe pectus excavatum (chest index 3.7) underwent MIRPE resulting in 100% correction of the chest wall abnormality. One year after MIRPE, the patient acquired a very pronounced pectus carinatum. Despite immediate removal of the pectus bar, the pectus carinatum was persistent for 2 months. The complication of pectus carinatum 1 year following MIRPE is rare and presents unique management dilemmas. Careful postoperative follow-up after MIRPE is recommended to identify patients at risk for development of a carinatum-like deformity so that bar removal can be performed in a timely fashion to halt or reverse the protrusion process. INTRODUCTION
T
for repair of pectus excavatum (PE) has evolved significantly over the last 4 years since the introduction of the technique of minimally invasive repair of pectus excavatum (MIRPE), also known as the Nuss technique.1 The cosmetic results achieved with the MIRPE are considered superior to those of the conventional open operation originally described by Ravitch,2 and the popularity of MIRPE has grown exponentially among pediatric surgeons despite limited reports of outcomes analysis of this new operation. Common complications of MIRPE include bar displacement, pneumothorax, infection, and pleural effusions. Unusual complications include thoracic outlet syndrome, pericarditis, massive blood loss, cardiac dysrhythmias, cardiac injury, sternal erosion, and pseudoaneurysm formation.3 The purpose of this report is to describe a rare case of acquired pectus carinatum that developed after MIPRE. This is the first report identifying acquired pectus carinatum as a complication of MIRPE. A previously reported review of 251 cases of MIRPE3 did not find a single case of protrusion deformity following this operation. HE OPERATIVE TREATMEN T
Division of Pediatric Surgery Medical University of South Carolina, Charleston, South Carolina.
41
HEBRA ET AL.
CASE REPORT The patient was a healthy 13-year-old boy with a history of severe pectus excavatum (chest index 3.7) who underwent uncomplicated MIRPE with placement of a single pectus support bar. He immediately achieved complete correction of the chest wall deformity and was discharged home 5 days after the operation. A postoperative chest radiograph demonstrated good positioning of the bar without any displacement or angulation. The patient had an uneventful recovery and did not have any early complications. He had three postoperative visits at 3 weeks, 6 months, and 1 year. At his 1-year follow-up visit, he was noted to have developed a very pronounced symmetric protrusion of the body of the sternum and costal cartilage with a lateral depression of the ribs consistent with severe pectus carinatum (Fig. 1). The stainless steel strut bar was immediately removed as an outpatient operation, but the acquired chondrosternal prominence did not improve (Fig. 2). He has been followed for 10 months since strut removal without any improvement of his acquired pectus carinatum.
DISCUSSION Pectus excavatum (also known as funnel chest) is a congenital chest wall deformity in which the sternum and adjoining cartilage are depressed within the chest cavity. The severity of this chest wall abnormality is cosmetically unappealing and in some patients may result in decreased exercise tolerance and adverse cardiopulmonary effects.4 The surgical management of PE has evolved to correction of using minimally invasive techniques. Specifically, in 1997, Dr. Nuss1 delineated the technique of minimally invasive repair of PE (MIRPE) that has been considered superior to the open approach,5 mainly because of minimal skin incisions, placement of a substernal strut without resection of cartilage, shorter operative time, reduced blood loss, decreased recovery time to full activity, and better cosmetic results.6 Consequently, the utilization of MIRPE has gained popularity among pediatric surgeons. As more patients undergo MIRPE, however, awareness of potential complications and outcomes became very important. In a review of 251 cases of MIRPE,4 the early complications and problems were delineated. The most common complication identified was pectus bar displacement/rotation (9.2%) followed by pneumothorax (4.8%), infection (2%), pleural
FIG. 1. Appearance of chest 1 year after minimally invasive repair of pectus excavatum. Note pronounced symmetric protrusion of body of sternum and costal cartilage with lateral depression of ribs consistent with acquired pectus carinatum chest wall deformity.
42
PECTUS CARINATUM AFTER EXCAVATUM REPAIR
FIG. 2. Stainless steel strut bar was immediately removed as outpatient operation, but acquired chondrosternal prominence did not improve.
effusion (2%), thoracic outlet obstruction (0.8%), cardiac injury (0.4%), sternal erosion (0.4%), pericarditis (0.4%), and thoracic artery pseudoaneurysm (0.4%). However, anterior protrusion of the sternum and ribs was not reported as a complication among the 251 patients reviewed. In the current report, we describe the occurrence of acquired pectus carinatum as a sequela of MIRPE. Our patient was a healthy 13-year-old boy who underwent uncomplicated MIRPE for severe pectus excavatum. He was followed for three postoperative visits, and at 12 months of follow-up, acquired pectus carinatum was identified. Immediate removal of the pectus bar did not change the acquired chest wall deformity, even at 10 months. To our knowledge, there is only one other report describing acquired pectus carinatum following MIRPE.7 In a review by Engum and associates,1 one patient believed to have a connective tissue disorder had a marked pectus carinatum defect identified 6 months postoperatively that necessitated removal of the pectus bar. A second patient in the report developed a mild carinatum defect and was followed closely to monitor the long-term appearance of the chest wall abnormality without bar removal. While unusual, these reports of acquired pectus carinatum following MIRPE illustrate the need for careful postoperative follow-up to help identify patients at risk for the development of an acquired carinatum deformity. Our patient was managed with immediate removal of the pectus bar in order to halt and, one hopes, reverse the protrusion process. To date, the acquired pectus carinatum remains unchanged without evidence of pectus excavatum recurrence. It is possible that removal of the bar prior to development of severe pectus carinatum could have prevented the occurrence of this problem, although it could have resulted in recurrence of the pectus excavatum. Thus, we believe that careful monitoring at 3-month intervals in the first year after MIRPE is important, as well as patient and family awareness of the possibility of this complication.
CONCLUSION As the popularity of MIRPE continues to increase, the indications for surgery, patient selection criteria, as well as complication management must be carefully reported and reviewed in order to optimize longterm outcomes. This report demonstrates the importance of establishing a national database to monitor technical variations, complications, and outcomes of this minimally invasive surgical technique. 43
HEBRA ET AL.
REFERENCES 1. Nuss D, Kelly Jr RE, Croitoru DP, et al. A 10-year review of a minimally invasive technique for the correction of pectus excavatum. J Pediatr Surg 1998;33:545–552. 2. Ravitch MM. The operative treatment of pectus excavatum. Ann Surg 1949;129:429–444. 3. Hebra A, Swoveland B, Egbert M, et al. Outcome analysis of minimally invasive repair of pectus excavatum: Review of 251 cases. J Pediatr Surg 2000;35:252–258. 4. Kowalewski J, Barcikowski S, Brocki M. Cardiorespiratory function before and after operation for pectus excavatum: Medium term results. Eur J Cardiothorac Surg 1999;13:275–279. 5. Robicsek F, Fokin A. Surgical correction of pectus excavatum and carinatum. J Cardiovasc Surg 1999;40:725–731. 6. Hebra A. Minimally invasive pectus surgery. Chest Surg Clin North Am 2000;10:1–9. 7. Engum S, Rescorla F, West K, Rouse T, et al. Is the grass greener? Early results of the Nuss procedure. J Pediatr Surg 2000;35:246–251.
Address for reprint requests to: André Hebra, M.D. Director Minimally Invasive Pediatric Surgery Program All Children’s Hospital 880 6th Street S. Suite 210 St. Petersburg, FL 33701 E-mail:
[email protected]
44
