ARTICLE IN PRESS Journal of Cranio-Maxillofacial Surgery (2006) 34, 107–112 r 2005 European Association for Cranio-Maxillofacial Surgery doi:10.1016/j.jcms.2005.08.009, available online at http://www.sciencedirect.com
Case Report Atypical cyst formation following chin augmentation using a nasal osteocartilaginous graft Frank LAZAR1, Axel zur HAUSEN2, Robert MISCHKOWSKI1, Joachim E. ZO¨LLER1 1
Department for Oral and Craniomaxillofacial Plastic Surgery (Head: Professor Dr. Dr. J.E. Zo¨ller); Department of Pathology (Head: Professor Dr. H.P. Dienes), University of Cologne, Germany
2
The purpose of this paper is to present a case of an intraosseous mucocele that was detected 5 years after chin augmentation using a nasal osteocartilaginous graft. Although complications of this technique have been described as negligible, the surgeon must be aware of possible long-term side effects and should remove all nasal mucosa from the graft prior to implantation. r 2005 European Association for Cranio-Maxillofacial Surgery
SUMMARY.
Keywords: cyst; chin; augmentation; nasal; graft
sub-mental region. The symptoms had been present for at least 6 weeks prior to referral. The past medical history included a rhinoplasty in 1996 (at the age of 18) that had been combined with a mentoplasty using the specimen (nasal hump) and placing it into an epiperiosteal fold through a submental incision. The postoperative course was uneventful (Fig. 1). But since January 2002 he had experienced infections in the chin region. Clinical examination revealed an abscess, explaining the patient’s complaints. Vitality tests of the anterior lower teeth were normal with no evidence of either periodontal disease or caries. A panoramic radiograph (Fig. 3) showed an ovoid apical radiolucency with no anatomical relationship to the lower incisors. It had a well-defined margin and no signs of a perforation of the lower border of the mandible. In the orthopan tomogram taken 1996 (prior to chin augmentation), no pathological lesion can be seen (Figs. 2 and 3). A provisional diagnosis of an atypical cyst with microbial superinvasion was made and exploration under local anaesthesia followed after mucosal incision. Biopsy was performed as a cystostomy with a long-term intraoral drainage following (Fig. 4). Histological examination revealed an implantation cyst lined by respiratory epithelium (Fig. 5). Postoperatively the radiological controls showed diminution of the cystic lesion but without complete osseous replacement within 5 months. Thus it was decided to perform a cystectomy under general anaesthesia combined with an osteoplasty via an intraoral approach using an iliac crest graft.
OBJECTIVES There are various procedures to augment the chin, such as using alloplastic implants (Friedland et al., 1976; Feuerstein, 1978; Flowers, 1991; Harada et al., 1993; Gubisch and Kotzur, 1998), chin osteotomies and grafting techniques with costal bone or nasal cartilage grafts (Hofer, 1942; Trauner and Obwegeser, 1957; Koele, 1961; Safian, 1966; Adams, 1987; Davis, 1983; Stambaugh, 1992; Allcroft et al., 1994; Bujia, 1994; Glasgold and Glasgold, 1994; Converse, 1950; Karacaoglan et al., 1998; Karras and Wolford, 1998; Choe, 2000; Mottura, 2002). The use of the nasal hump and septal cartilage has already been described by Aufricht (1934, 1958). In the latter article (1958), he reported 700 cases with good clinical results without observing resorption. Although others also reported similar and favourable results when using this technique, Anastassov and Lee (1999) as well as Imholte and Schwartz (2001) described the development of respiratory mucoceles in the chin region as a major complication after nasal hump transplantation. The case presented here is another one of the rare manifestations of a respiratory implantation cyst, the origin of which was probably based on epithelial cells that had been incompletely removed from the resected nasal hump. PATIENT A 24-year-old male patient complained of discomfort, slight swelling, reddening and pain affecting the 107
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Fig. 1 – Pre- (A: 10/1996, 18 y/o male) and postoperative (B: 9/2002, 24 y/o male) lateral head X-rays prior to and after removal of a nasal hump and genioplasty.
Fig 2 – Orthopantomogram, October 1996, prior to genioplasty and rhinoplasty, no evidence of pathological osseous lesions.
Fig. 3 – Orthopantomogram, January 2002, radiolucent lesion found in the anterior mandible with a circular radiodense lining.
Similar to the observations described by Imholte and Schwartz (2001) the lesion in this case was found to be adherent to the remnants of the transplanted cartilaginous graft (Fig. 6) anterior to the mandibular symphysis. There was also osteolysis of the anterior cortex. Dissection of the cystic soft tissue was carried out completely although the cavity turned out to be multilocular. Additional genioplasty was carried out by transplanting one layer of cortical bone (inner cortex of the iliac crest) to augment the chin (Fig. 7).
The remaining course has been uneventful showing a complete osseous regeneration of the cavity and a pleasing restoration of the chin contour over a follow-up period of 212 years (Fig. 8). HISTOLOGY The tissue obtained was fixed in 4.5% phosphatebuffered formaldehyde (pH 7), and embedded in paraffin wax for light microscopy. Four-micrometer
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Fig. 4 – Excisional biopsy performed as a cystostomy followed by long term intraoral drainage.
Fig. 5 – Microscopical specimen, HE, 25, respiratory mucocoele consisting of cartilaginous wall lined by a thin, regular respiratory epithelium, compatible with a so-called ‘implantation cyst’ (Pathologisches Institut der Universita¨t zu Ko¨ln, Dr. A. zur Hausen).
sections were stained with haematoxylin and eosin. The initial biopsy as well as the final specimen from cystectomy revealed identical histological appearances. A respiratory mucocoele consisting of a cartilaginous wall lined by a thin, regular respiratory epithelium, compatible with a so-called implantation cyst (Fig. 5). DISCUSSION Genioplasty is a reliable technique in maxillofacial plastic surgery. Sliding chin osteotomy is common in maxillofacial surgery (Hofer, 1942; Trauner and Obwegeser, 1957; Koele, 1961) avoiding a donor site and offering proper biological behaviour. However,
this method requires a well-trained surgeon with some degree of experience as stated by Mottura (2002). Alternatively, autologous grafts can be used. Common donor sites are iliac crest and rib. The additional surgical intervention at the donor site and their inherent morbidity are disadvantages. Alloplasts avoid additional surgical interventions. Silicone or ceramics are used widely (Feuerstein, 1978; Flowers, 1991; Karras and Wolford, 1998) and the surgical approaches are either transoral or transcutaneous (Allcroft et al., 1994). Complications arising after implantation of alloplasts are as common as with other techniques for genioplasty (Friedland et al., 1976; Matarasso et al., 1996; Zide, 1999).
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Fig. 6 – Intraoperative view, multilocular appearance following cystectomy.
Fig. 7 – Osseous defect filled and chin contour augmented using a cortical iliac graft fixed with bone screws.
Genioplasty incorporates specific side effects that have been reported to have a frequency between 0% and 2%: dislocation, infection, neurosensory disturbances or resorption are noted independently of the surgical method and approach or the material used. There is presently no ‘holy grail’ method for genioplasty (Tresley et al., 1972; Vedtofte et al., 1991). Mottura (2002) and Karacaoglan et al. (1998) recently described minor chin augmentations using the same transcutaneous, extraoral submental approach as had been described by Aufricht (1934). Subsequent preparation and placement of the cartilaginous transplant into a subperiosteal (Mottura, 2002) or epiperiosteal pocket followed. No side effects nor postoperative long-term complications were reported. Resorption of the graft was reported to be minimal although radiological studies 6 months postoperatively by Karacaoglan et al. (1998) revealed the remaining portion of the graft being not more
than 75% of the initial volume. Mottura (2002) reported 10 cases with a follow-up period of 3–8 years and an absence of resorption of the graft or alteration of the recipient bone. Aufricht (1934) and Karacaoglan et al. (1998) described a supraperiosteal placement whereas Mottura preferred a subperiosteal placement. In the case presented here an osteocartilagineous chin implant placed subperiosteally led to the development of an atypical intraosseous mucocoele 5 years postoperatively. Therefore therapy consisted in cystostomy, long-term drainage and subsequent cystectomy combined with osteoplasty. In contrast to other authors, a multilocular cystic lesion (Figs. 4–6) was found. It was difficult to dissect the cystic lesion from the bone cavities. Consequently, only microscopical fragments can be demonstrated. There were only two case presentations (Anastassov and Lee, 1999; Imholte and Schwartz, 2001) reporting mucocoeles which had grown from respiratory epithelium
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Fig. 8 – Result 4-months postoperatively.
as a possible consequence of persistent mucosa on the graft’s surface. In the past, some authors discussed negative effects of persistent nasal mucosa on the tissue of the recipient site (Gorlin, 1957; Piecuch et al., 1980). The majority of authors thus recommend separation of all the adherent soft tissues from the transplanted bone/cartilage. Therefore, Mottura (2002) recommended and described in detail a microscopically assisted, rigorous removal of the remaining portion of adherent nasal mucosa.
CONCLUSION The case presented underlines the potential hazard of uncontrolled cell growth when epithelial cells are accidentally transplanted to another environment within the human body. A so-called implantation cyst may result. Therefore, it must be emphasized that a transplant taken from the nasal hump incorporates some rare but serious risks that can be
avoided by removal of all adherent nasal mucosa from the graft.
References Adams JS: Grafts and implants in nasal and chin augmentation. Otolaryngol Clin North Am 20: 913–930, 1987 Allcroft RA, Friedman CD, Quatela VC: Cartilage grafts for the head and neck augmentation and reconstruction. Otolaryngol Clin North Am 27: 69–80, 1994 Anastassov GE, Lee H: Respiratory mucocoele formation after augmentation genioplasty with nasal osteocartilaginous graft. J Oral Maxillofac Surg 57: 1263–1265, 1999 Aufricht G: Combined nasal plastic and chin plastic: correction of microgenia by osteocartilaginous transplant from large hump nose. Am J Surg 25: 292–296, 1934 Aufricht G: Combined plastic surgery of the nose and chin. Am J Surg 95: 237–238, 1958 Bujia J: Determination of the viability of crushed cartilage grafts: clinical implications for wound healing in nasal surgery. Ann Plast Surg 32: 261–265, 1994 Choe KS: Chin augmentation. Facial Plast Surg 16: 45–54, 2000 Converse JM: Restoration of the facial contour by bone grafts introduced through the oral cavity. Plast Reconstr Surg 6: 295–300, 1950
ARTICLE IN PRESS 112 Journal of Cranio-Maxillofacial Surgery Davis PKB: Chin augmentation with rhinoplasty: a tutorial dissertation. Br J Plast Surg 36: 204–209, 1983 Feuerstein SS: Intraoral augmentation mentoplasty with a hinged silastic implant. Arch Otolaryngol 104: 383–387, 1978 Flowers SR: Alloplastic augmentation of the anterior mandible. Clin Plast Surg 18: 107–138, 1991 Friedland JA, Coccaro PJ, Converse JM: Retrospective cephalometric analysis of mandibular bone absorption under silicon rubber chin implants. Plast Reconstr Surg 57: 144–151, 1976 Glasgold AI, Glasgold MJ: Intraoperative custom contouring of the mandible. Arch Otolaryngol Head Neck Surg 120: 180–184, 1994 Gorlin RJ: Potentialities of oral epithelium manifest by mandibular dentigerous cysts. Oral Surg 10: 271–284, 1957 Gubisch W, Kotzur A: Our experience with silicone in rhinomentoplasty. Aesth Plast Surg 22: 237–244, 1998 Harada K, Torikai K, Funaki J: Augmentation genioplasty with hydroxyapatite blocks. Int J Oral Maxillofac Surg 22: 265–266, 1993 Hofer O: Die operative Behandlung der alveola¨ren Retraktion des Unterkiefers und ihre Anwendungsmo¨glichkeit fu¨r Prognathie und Mikrogenie. Dtsch Zahn Mund Kieferheilkd 9: 121–132, 1942 Imholte M, Schwartz HC: Respiratory implantation cyst of the mandible after chin augmentation: report of case. Otolaryngol Head Neck Surg 124: 586–587, 2001 Karacaoglan N, Akbas H, Eroglu L, Incesu L: Chin augmentation using diced cartilage. Eur J Plast Surg 21: 254–256, 1998 Karras SC, Wolford LM: Augmentation genioplasty with hard tissue replacement implants. J Oral Maxillofac Surg 56: 549–552, 1998 Koele H: Zur operativen Behandlung der Progenie. O¨sterr Z Stomatol 58: 25–29, 1961 Matarasso A, Elias AC, Elias RL: Labial incompetence: a marker for progressive bone resorption in silastic chin augmentation. Plast Reconstr Surg 98: 1007–1014, 1996
Mottura AA: Chin augmentation with nasal osteocartilaginous graft. Plast Reconstr Surg 109: 783–787, 2002 Piecuch JF, Eisenberg E, Segal D, Carlson R: Respiratory epithelium as an integral part of an odontogenic keratocyst: report of case. J Oral Surg 38: 445–447, 1980 Safian J: Progress in nasal and chin augmentation. Plast Reconstr Surg 37: 446–452, 1966 Stambaugh KI: Chin augmentation. Arch Otolaryngol Head Neck Surg 118: 682–686, 1992 Trauner R, Obwegeser H: The surgical correction of mandibular prognathism and retrognathia with consideration of genioplasty. Oral Surg Oral Med Oral Pathol 10: 677–689, 1957 Tresley IJ, Kaufinamm Arenberg I, Polterock J: Augmentation mentoplasty—reflections. Laryngoscope 82: 2092–2102, 1972 Vedtofte P, Nattestad A, Hjo¨rting-Hansen E, Svendssen H: Bone resorption after advancement genioplasty. J Cranio Max Fac Surg 19: 102–107, 1991 Zide BM: Chin surgery, 1: augmentation—the allures and the alerts. Plast Reconstr Surg 104: 1843–1853, 1999
Dr. med. Frank C. LAZAR Klinik und Poliklinik fu¨r Zahna¨rztliche Chirurgie und fu¨r Plastische Mund-, Kiefer- und Gesichtschirurgie Klinikum der Universita¨t zu Ko¨ln Kerpener Strasse 62 50937 Ko¨ln Germany Tel.: +49 221 478 5775 (secretary),/5793 (office) Fax: +49 221 478 5774 E-mail:
[email protected],
[email protected] Paper received 22 February 2005 Accepted 18 August 2005