Pectus Excavatum Reconstruction With Silicone Implants. Long-Term Results and a Review of the English-Language Literature

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1 REVIEW ARTICLES Pectus Excavatum Reconstruction With Silicone Implants Long-Term Results and a Review of the English-Language Literature Bart Jorrit Snel, MD,* Cees A. Spronk, MD, Paul M. N. Werker, MD, PhD,* and Berend van der Lei, MD, PhD* Abstract: Pectus excavatum, or funnel chest, is an anterior chest wall deformity, characterized by a depressed sternum. Frequently used techniques for pectus excavatum repair are extensive thoracic surgical corrections at a young age. At an adult age, custom-made silicone implants can be used as a less extensive surgical option. Thus far, there are only a limited number of reports in the English-language literature concerning this treatment modality. We evaluated our series of 16 treated with custom-made silicone implants, assessing complications, patient satisfaction, and aesthetic outcome. Seven (44%) had complications, mostly seroma formation (31%). Ten of 12 (83%), who were evaluated at the long-term, reported improved patient satisfaction and 9 of 13 (69%), in whom aesthetic results were assessed, had a good or excellent aesthetic outcome. Our study, combined with a review of the English-language literature, demonstrates that there is a place for custom-made silicone implants in the treatment of pectus excavatum, especially in less severe cases at an adult age. Preoperative counseling with emphasis on the likelihood of seroma formation is advocated. Key Words: pectus excavatum, silicone, implants, endoprosthesis (Ann Plast Surg 2009;62: ) Pectus excavatum (PE), or funnel chest, is one of the most common congenital anomalies. It is described as an anterior chest wall deformity, characterized by a depressed sternum. This deformity is approximately 6 times more common than pectus carinatum. It occurs in 1 in every 400 to 1000 births. 1,2 Men are afflicted approximately 5 times more often than women. 3 The condition is often first recognized during infancy. Slowly it becomes more pronounced and with the rapid skeletal growth of adolescence, almost all experience a marked increase in severity. After bone maturation has been achieved, the deformity usually remains constant throughout adult life. Untreated PE may often lead to embarrassment and affects the psychosocial development. 4 The most frequently used techniques for PE repair are extensive thoracic surgical corrections at an age of 12 to 16 years. 5,6 However, it is common for some not to seek surgical correction until at an adult age. At this age, a less extensive surgical option is the use of a custom-made silicone implant to Received October 26, 2007, and accepted for publication, after revision, April 16, From the *Department of Plastic Surgery, University Medical Center Groningen, Groningen; Department of Plastic, Reconstructive, Aesthetic, and Hand Surgery, Medical Center of Leeuwarden, Leeuwarden; and Private Clinic Heerenveen, Heerenveen, The Netherlands. Reprints: Berend van der Lei, MD, PhD, University Medical Center Groningen, Hanzeplein 1, 8934 AD Groningen, The Netherlands. b.van.der.lei@ plchir.umcg.nl. or heslei@planet.nl. Copyright 2009 by Lippincott Williams & Wilkins ISSN: /09/ DOI: /SAP.0b013e31817d878c improve appearance. This treatment modality can be valuable and relatively simple, especially in cases without respiratory or cardiologic symptoms. Thus far, there is only a limited number of reports in the English-language literature concerning long-term evaluation of PE with silicone implants (Table 1). Therefore, in this report we would like to share our experience with a series of 16, and discuss our results and complications of this treatment modality together with a review of the English-language literature. PATIENTS AND METHODS Patient Characteristics Between 1987 and 2007, 16 were treated for PE by means of a custom-made silicone implant (for detailed patient characteristics, see Table 2). There were 14 men and 2 women. At the moment of operation, their mean age was 43 years, ranging from 21 to 64 years. Mean follow-up was 6 years, with a range of 6 months to 20 years. None of the reported any respiratory or cardiologic impairment. One patient had a Ravitch procedure 5 10 years earlier and was treated with a silicone implant for further improvement of the cosmetic result (case 16). Another patient had received a dermis fat graft 24 years before consulting us and an implant of 60% acrylate cellulosis 2 years thereafter, before she was treated with a silicone implant (case 12). Manufacturing of Implant and Surgical Technique In the preoperative period, a custom-made silicone implant was manufactured, preformed to fit precisely into the pectus cavity of the patient. This was achieved by modeling a cast in exact correspondence to the impression of the chest wall, which was used as a template for the final implant. Surgery was performed according to a standardized procedure under general anesthesia: first, by means of a horizontal subxiphoid skin incision, a subcutaneous pocket was prepared over the sternum. Then, the custom-made silicone implant was inserted, followed by closure of the wound in layers over a suction drain. Outcome Analysis All medical charts were reviewed for analysis of the operation, complications, and clinical course. In addition, we were able to contact 12 for extensive patient evaluation. Patient satisfaction was analyzed using a PE evaluation questionnaire, which is a modification of the Breast Evaluation Questionnaire, designed by Anderson. 7 With this questionnaire, the impact of the PE deformity on body image of the patient can be scored. Moreover, additional information was obtained concerning the subjective improvement after surgery, as well as of the social impairment caused by the deformity before the operation and at the moment of long-term follow-up. The aesthetic outcome was analyzed by scoring the postoperative result as defined by E. Strasser, based upon photographs taken in the early postoperative period and at the long-term evalu- Annals of Plastic Surgery Volume 62, Number 2, February

2 Snel et al Annals of Plastic Surgery Volume 62, Number 2, February 2009 TABLE 1. Review of the English-Language Literature Yr Journal Pts Cmp Fup (yr) Patient Satisfaction Aesthetic Outcome Nordquist et al Scand J Plast Reconstr 27* 13 6 Improvement in 11 Surg Hand Surg Wechselberger et al Ann Plast Surg ,5 Improvement in 17 Excellent good results in 18 Margulis et al J Plast Reconstr Aesth 7 0 1,4 Excellent, 1 patient show Surg Horch et al Aesth Plast Surg Improvement in all Snel et al Ann Plast Surg 16* 7 6 Improvement in 11/12 Excellent good results in 9/13 *Nordquist et al, reported of a series of 27, but patient satisfaction was assessed in only 19. In our series of 16, patient satisfaction was assessed in 12 ; aesthetic outcome was assessed in 13. Pts indicates no. in each series; Cmp, percentage of reported postoperative complications; Fup, follow-up. TABLE 2. Patient Characteristics, Patient Satisfaction, and Aesthetic Outcome Case No. Sex Age (yr)* Fup (yr) Complication Intervention Preop ( 6 to 6) Postop ( 6 to 6) Anderson (0 5) Strasser 1 F Seroma, luxation Reoperation Good 2 M 35 1 Seroma Explantation Good 3 M 20 1 Seroma, luxation Reoperation Mediocre 4 M ,2 Mediocre 5 M 25 10,5 Discomfort 4 Reoperations 2 3 3,4 Good 6 M 43 10, ,5 Good 7 M ,3 Good 8 M 43 6, ,6 Excellent 9 M 21 6,5 10 M 23 6 Seroma Aspiration 3 1 4,4 Mediocre 11 M ,5 Good 12 F 64 4 Pain, luxation Explantation M 21 0,5 14 M 28 3,5 Show 4 4 1,8 Poor 15 M 24 3, Good 16 M 26 3 Seroma Aspiration Good *The reported ages are at time of the first operation. Fup indicates follow-up; Preop and Postop indicate patient satisfaction rates preoperatively and postoperatively, respectively, ranging from 6 (very unsatisfied) to 6 (very satisfied). ation moment. 8 None of the reported any change in the appearance of their chest at any time in the postoperative period. The Strasser system divides aesthetic outcome into 5 categories: malposition, distortion, asymmetry, contour deformity, and scar. Grades 1 to 5 are assigned to each category and these add up to a final result, being either excellent, good, mediocre, or poor. RESULTS Complications (Table 2) In 7 of 16 (43%), there were complications in the early postoperative period. In 5 (31%), there was seroma production for a prolonged period. This resulted in explantation of the implant in one case (case 2) after 5 months, because of never ending extreme seroma production. This patient was subsequently treated by means of a Rehbein thoracic surgical procedure. 9 In 2 other (cases 1 and 3) seroma formation with concomitant implant dislocation was managed with a reoperation and repositioning of the implant. In the other 2 (cases 10 and 16) the 206 seroma formation could successfully be managed by means of repetitive puncture evacuation in a period of 3 months. In 1 patient (case 12) the implant was explanted because of persisting pain and implant malposition. This patient was subsequently treated with tricalcium phosphate scaffolds to fill the pectus cavity. 10 Another patient (case 5) underwent 4 reoperations because of persisting discomfort. Patient Satisfaction Results derived from the questionnaires are reported in Table 2. The preop and postoperative scores reflect patient satisfaction and the Anderson score is a representation of the body image. Eleven of the 12 evaluated (92%) reported significant postoperative improvement. This corresponded to the reported patient bodyimage scores: 10 had scores above 4, indicating a positive body image in these. One patient (case 14) did not report postoperative improvement and had a low body image due to implant show. This also corresponded with a low aesthetic Strasser score Lippincott Williams & Wilkins

3 Annals of Plastic Surgery Volume 62, Number 2, February 2009 Pectus Excavatum Reconstruction FIGURE 1. Case 5. Note the obvious preoperative PE deformity and the good postoperative aesthetic result, as defined by the Strasser system, 6 years after implantation: there is no malposition of the implant and no distortion, asymmetry or contour deformity. The scar is just discernable. Aesthetic Outcome We were able to score the aesthetic result in 13 of 16, according to the Strasser system: there was one case with an excellent result (case 8 with no visible scar). In 8 the result was not excellent but good, because of a visible subxiphoid scar (Fig. 1). Three had a mediocre result either because of an obvious contour deformity, noticeable malposition, or distortion (Fig. 2) and 1 patient with a poor aesthetic outcome because of an obvious contour deformity, noticeable malposition, distortion, and asymmetry (case 14; Fig. 3). These findings corresponded with a low patient satisfaction score. DISCUSSION The long-term evaluation of custom-made silicone implants for the repair of PE in adults in this study demonstrates that satisfactory aesthetic results with a high patient satisfaction rate can be achieved with this procedure in the majority of cases, despite a rather high complication rate in the early postoperative period. Seroma formation is the most frequent complication and may result in all kinds of secondary complications such as chronic pain, discomfort, and implant dislocation. However, if the problem of seroma formation is solved (repetitive puncture evacuation in a period of 3 months) or if this complication does not occur at all, a stable, pleasing long-term aesthetic result can be achieved, leading to a high patient satisfaction rate. Thus far, since the introduction of silicone implants in the late 1970s 11,12 few studies have been published regarding the long-term evaluation of custom-made silicone implants for PE (Table 1). In 2001, Nordquist, Svensson, and Johnsson were the first to report their experience with PE repair with this treatment modality, presenting a series of Fifteen of the 19 (79%) that were evaluated from this series by means of a questionnaire, reported improvement of appearance and well-being. In 2001, Wechselberger et al also reported on a series of Using a nonspecific analysis, performed by 2 plastic surgeons, they reported that 80% of their showed excellent results, 10% demonstrated good results and 10% demonstrated acceptable results. In 2006, Margulis et al 15 reported excellent correction in a rather small series of 7, with a minimal implant show in FIGURE 2. Case 4, 10 years after implantation, illustrating an example of a mediocre result, as defined by the Strasser system: malposition and distortion of the implant is noticeable and the contour deformity is obvious Lippincott Williams & Wilkins 207

4 Snel et al Annals of Plastic Surgery Volume 62, Number 2, February 2009 FIGURE 3. Case 14, 3 years after implantation, demonstrating the only poor result in our series, as defined by the Strasser system: there is obvious malposition, distortion, and asymmetry of the implant, together with a disturbing contour deformity. just 1 patient. They pointed out that in with paucity of subcutaneous fat, there is a greater chance of occurrence of this complication. We also have experienced this problem of implant show in 1 patient (case 14) in our series of 16, who was very unsatisfied with the overall postoperative result. Patients, especially thin persons, should be well informed about the possibility of postoperative implant show. A lipofilling procedure preceding the implantation could perhaps be considered as a method to increase the thickness of the covering skin and subcutaneous area. In 2006 Horch et al 16 described just 5 who underwent PE repair with a custom-made silicone implant, among a series of 594 other who were treated with a Ravitch or Erlangen thoracic surgical procedure. All 5 were satisfied with the outcome as reported at the follow-up of examination, after 24 to 45 months. Evaluation of our series of 16 demonstrates similar satisfactory results as those found in literature: within our series, both the aesthetic outcome, as evaluated by means of the Strasser score, and the patient satisfaction rate, as evaluated by means of a specially developed PE questionnaire, were high. Seroma formation was the most frequently observed complication in the early postoperative period in our series (5 of , 31%). This complication rate is low compared with that reported by Wechselberger et al, who reported seroma formation in 13 of 20 and similar to that of Nordquist, Svensson, Johnsson, who reported seroma formation in 10 of ,14 Why does this seroma formation occur at such a high rate? Siliconeinduced tissue damage clinically shows a wide variability and, although an immunologic reaction is suspected in most cases, a conclusive model of pathogenesis is not yet available. 17 Thinness of the skin and subcutaneous tissue in the area of implantation might be a factor of influence. In cases of breast augmentation, even in a thin patient, there is hardly any seroma formation in the early postoperative period, probably due to a thicker skin and subcutaneous tissue coverage of the implant. 18 The size of the implant might also be a factor contributing to this phenomenon. Despite the rather high complication rate after the use of custom-made silicone implants for PE, ultimately highly satisfactory results can be achieved in 83% of the cases. According to Strasser, in 9 of 13 (69%) an excellent or good result was obtained, whereas in 2 the procedure led to a mediocre result. One patient had a poor aesthetic outcome with severe implant distortion and show. This latter patient with a severe PE deformity in retrospect would have been a better candidate for thoracic surgical correction at a young age. Analyzing our results thoroughly, it was quite striking that the patient satisfaction rate was even higher than the aesthetic outcome scores: in most significant postoperative improvement of patient satisfaction was achieved. One could argue that patient satisfaction is the ultimately most important factor in outcome analysis. Even higher satisfaction rates might be achieved by improved patient education and subsequently lowering the aesthetic expectations. Based upon this long-term evaluation of 16 (Table 2), combined with a review of the English-language literature (Table 1), we can conclude that there is a definitive place for custom-made silicone implants as a treatment modality for PE., especially in adult without pulmonary or cardiologic disease. It definitely improves patient satisfaction and well-being. However, a rather high rate of seroma formation in the early postoperative period should be considered and in case of with little subcutaneous fat, there is a high likeliness of implant show. Lipofilling of the skin and subcutaneous tissues in the area of the defect, preceding the implantation of the custom-made silicone implant, might be a solution for both these potential complications. ACKNOWLEDGMENTS The authors thank B. Tebbes, medical photographer of the UMCG, for the photography. REFERENCES 1. Kelly RE, Shamberger RC, Mellins RB, et al. Prospective multicenter study of surgical correction of pectus excavatum: design, perioperative complications, pain, and baseline pulmonary function facilitated by internet-based data collection. J Am Coll Surg. 2007;205: Fonkalsrud EW. Current management of pectus excavatum. World J Surg. 2003;27: Shamberger RC. Congenital chest wall deformities. Curr Prob Surg. 1996; 33: Einsiedel E, Clausner R. Funnel chest: psychological and psychosomatic aspects in children, youngsters and young-adults. J Cardiovasc Surg. 1999; 40: Ravitch MM. The operative treatment of pectus excavatum. Ann Surg. 1949;122: Nuss D, Kelly RE Jr, Croitoru DP, et al. A 10-year review of a minimally invasive technique for the correction of pectus excavatum. J Pediatr Surg. 1998;33: Lippincott Williams & Wilkins

5 Annals of Plastic Surgery Volume 62, Number 2, February 2009 Pectus Excavatum Reconstruction 7. Anderson RC, Cunningham MD, Tafesse E, et al. Validation of the breast evaluation questionnaire for use with breast surgery. Plast Reconstr Surg. 2006;116: Strasser EJ. An objective grading system for the evaluation of cosmetic surgical results. Plast Reconstr Surg. 1999;104: Rehbein F, Wernicke HH. The operative treatment of the funnel chest. Arch Dis Child. 1957;32: Hench LL. Bioceramics: from concept to clinic. J Am Ceram Soc. 1991;74: Mendelson B, Masson JK. Silicone implants for contour deformities of the trunk. Plast Reconstr Surg. 1977;59: Lavey E, Apfelberg DB, Lash H, et al. Customized silicone implants of the breast and chest. Plast Reconstr Surg. 1982;69: Nordquist J, Svensson H, Johnsson M. Silastic implant for reconstruction of pectus excavatum: an update. Scand J Plast Reconstr Surg Hand Surg. 2001;35: Wechselberger G, Ohlbauer M, Haslinger J, et al. Silicone implant correction of pectus excavatum. Ann Plast Surg. 2001;47: Margulis A, Sela M, Neuman R, et al. Reconstruction of pectus excavatum with silicone implants. J Plast Reconstr Aesthet Surg. 2006;59: Horch RE, Stoelben E, Carbon R, et al. Pectus excavatum breast and chest deformity: indications for aesthetic plastic surgery versus thoracic surgery in a multicenter experience. Aesthetic Plast Surg. 2006;30: Winkler PA, Herzog C, Weiler C, et al. Foreign-body reaction to silastic burr-hole covers with seroma formation: case report and review of the literature. Pathol Res Pract. 2000;196: Siggelkow W, Klosterhalfen B, Klinge U, et al. Analysis of local complications following explantation of silicone breast implants. Breast. 2004;13: Lippincott Williams & Wilkins 209

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