- Email: [email protected]
Computer-assisted midface reconstruction in Treacher Collins syndrome part 2: Soft tissue reconstruction
Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680
Contents lists available at SciVerse ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Computer-assisted midface reconstruction in Treacher Collins syndrome part 2: Soft tissue reconstruction Christian Herlin a, *, Jean Charles Doucet a, b, Michèle Bigorre a, Guillaume Captier a, c a
Department of Plastic and Craniofacial Pediatric Surgery (Head: Pr Guillaume Captier), Faculty of Medicine, University of Montpellier, 371 Avenue du doyen Gaston Giraud, 34295 Montpellier Cedex 5, France b Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Dalhousie University, Halifax, Nova Scotia, Canada c Laboratory of Anatomy, Faculty of Medicine, University of Montpellier, France
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 10 June 2012 Accepted 4 January 2013
Introduction: Treacher Collins syndrome (TCS) midfacial involvement associate a skeletal hypoplasia centred on the zygoma to a hypoplasia of all surrounding soft tissues layers and an inferolateral lower lid pseudocoloboma. TCS soft tissue hypoplasia, which has not been well studied, continues to bring challenges in both the indication of surgical treatment and the prediction of their results. Material and method: From a standard magnetic resonance imaging (MRI) acquisition, we studied qualitatively and quantitatively the prezygomatic fat compartments and the buccal fat pad of two individuals with TCS whose age were 10 and 14 years. In parallel, we studied 20 controls at the same age to obtain a morphometric database of reference and compare our results. TCS soft tissue involvement was correlated to the results of our prior skeletal involvement study. Results: The midfacial fat compartments in TCS are severely hypoplastic, especially in the superficial and lateral compartments of the face (all P’s < 0.001). No significant correlation existed between the soft tissue and the skeletal involvement. Conclusions: To our knowledge, this is the first published study of TCS midfacial fat compartments. Their hypoplasia is an important part of the syndrome’s facial deformity. The knowledge of their anatomy, organization and volumetric variation is essential. Their re-establishment is key in the early treatment phases of this syndrome. Using the preoperative data, the morphometric database of reference, and surgical simulation, an appropriate surgical technique, going from an autologous fat graft to a free flap, can then be chose. Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Treacher Collins syndrome Franceschetti syndrome Soft tissue Lipofilling Fat Computer-assisted surgery Fat grafting Presurgical simulation
1. Introduction Treacher Collins syndrome (TCS) is a rare and severe craniofacial malformation with bilateral midfacial zygomatico-orbital bony involvement. The skeletal hypoplasia is centred on the zygoma, and is associated with hypoplasia of all the surrounding soft tissues layers, and an inferolateral lower lid pseudocoloboma. This soft tissue involvement presents an important aesthetic and reconstructive challenge, but remains poorly studied. The purpose of our study was to evaluate the facial fat compartments in TCS, using magnetic resonance imaging (MRI). Our specific aim was to evaluate which compartments are most affected, and in what proportion, by comparing them to a control population. This will help to guide the reconstruction and to select * Corresponding author. Tel.: þ33 (0)619715557; fax: þ33 (0)467603639. E-mail address: [email protected] (C. Herlin).
between different surgical techniques including alloplastic implants, dermal-fat graft, free flaps and autologous fat graft. 2. Materials and methods In parallel with our skeletal study, an MRI study was performed in two TCS patients aged 10 and 14 years. These patients were also evaluated with computed tomography (CT) to correlate the soft tissue and the skeletal involvement. To draw conclusions on the data acquired from the MRI and produce a morphometric database, 10 controls per age category were studied. The data studied came from 20 patients (40 “healthy” control sides) who had a craniofacial MRI for problems not related to the facial soft tissues. This database was also compared to the rare data found in the literature (Gosain et al., 2005; Loukas et al., 2006). A single examiner performed the data collection and analysis. All the MRI’s were acquired by the same machine (Philips Brilliance,
1010-5182/$ e see front matter Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jcms.2013.01.008
C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680
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Table 1 Buccal fat pad volumes (cm3) in TCS patients and in 10 controls per age category.
Fig. 1. Example of fatty ROI segmentation on MRI. Each ROI are represented by a colour (Buccal fat pads in yellow and green; Prezygomatic fat compartments in pink and orange).
1.5T). The smallest possible field of view was chosen to increase special resolution. T1 sequencing was performed in the axial, coronal and sagittal plane. T2 sequencing was only performed in the axial plane. Since the fat compartments were our regions of interest (ROI), no other sequences were chosen. Slice thickness was set at 1.5 mm to avoid a “dead space” effect while ensuring adequate resolution for the volumetric reconstruction. To study all prezygomatic fat compartments, the slices were realigned along a plane extending from the lateral canthus to the oral commissure, as advocated by Gosain et al., (2005). Post-image processing was realized with the MYRIANÒ expert v. 1.5.2 software (Intrasense SAS, Montpellier, France). The first step of soft tissue segmentation (Fig. 1) was carried out automatically. After manual verification, following the anatomical fat compartments description of Rohrich and Pessa (2007) and Aiache and Ramirez (1995), the following geometric parameters were determined: the buccal fat pad (BFP) volume, and the superficial and
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L
R
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Treacher Collins Control 1 Control 2 Control 3 Control 4 Control 5 Control 6 Control 7 Control 8 Control 9 Control 10
4.38 6.217 5.788 7.26 6.356 5.661 6.124 5.741 6.143 7.223 6.123
4.355 7.115 6.019 6.983 6.424 8.826 6.342 5.12 6.23 7.124 6.294
6.96 10.9 9.887 7.436 10.3 8.672 9.214 8.876 8.812 8.945 8.786
6.859 11 9.873 7.667 10.105 7.923 9.245 8.245 8.123 8.751 8.642
deep prezygomatic fat volumes (PZF). The prezygomatic fat volumes included the medial, middle and lateral temporal cheek fat compartments, the suborbicularis compartment, and the retroorbicularis fat compartment (Rohrich and Pessa, 2007). The volume measurement of each individual prezygomatic compartment was not possible due to the MRI resolution. The soft tissue deficiency was then compared to the skeletal deficiency found on CT, to calculate a volumetric ratio (bone vs fat) (Fig. 2). All statistics were computed with the SPSS Statistics 17.0 software (version 17.0.0, SPSS Inc, Chicago, IL). Appropriate bivariate and multivariate statistics were performed, and the level of statistical significance was set at P < 0.05. 3. Results 3.1. Fat compartments study The TCS buccal fat pad volumes found on MRI were significantly lower than the volumes found in the control population (P < 0.001) (Table 1 and Fig. 2). The control population measurements were similar to the literature data (4e6). In TCS the superficial and deep prezygomatic fat volumes were also significantly lower than the control population volumes (P < 0.001) (Table 2 and Fig. 3). The superficial fat compartments were proportionally more affected towards the orbital rim. The deficit gradient of the superficial fat compartments is shown schematically in Fig. 4. In the facial “cleft” area of TCS patients the deep fat compartments persisted. When focussing on the BFP, the temporal and masseteric extensions were more affected. Subjectively, the compartmentalization of the PZF was also increased and the fibrous tract defined as soft tissue ligaments (Moss et al., 2000; Rohrich and Pessa, 2007), which are sometimes visible on MRI, seemed more numerous and less structured in the syndromic patients.
Table 2 Prezygomatic fat volumes (cm3) in TCS patients and in 10 controls per age category.
Fig. 2. Buccal fat pad volumes (cm3) in TCS patients and in 10 controls per age category.
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L
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L
Treacher Collins Control 1 Control 2 Control 3 Control 4 Control 5 Control 6 Control 7 Control 8 Control 9 Control 10
7.267 13.9 12.4 13.3 14.0 12.5 14.7 13.1 13.3 12.9 13.8
7.252 14.5 13.2 12.2 13.6 12.9 14.3 12.9 13.1 12.7 13.5
10.8 19.0 22.3 17.7 20 14.9 18.1 22.1 17.3 20.2 20.8
11.4 19.3 21.9 18.1 19.7 15.3 17.6 21.6 17.1 20.4 21.1
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C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680 Table 3 Percentage of volume deficiency compared to the controls and comparison of the skeletal and soft tissue involvement in TCS patients.
Orbit Zygoma PZF BFP
Patient 1
Patient 2
Patient 3
Patient 4
3.6% 62.1% 45.6% 31.9%
4.9% 81.2% 42.3% 20.8%
15% 97.9% X X
0% 98.9% X X
PZF: Prezygomatic fat; BFP: Buccal fat pad.
Fig. 3. Prezygomatic fat volumes (cm3) in TCS patients and in 10 controls per age category.
3.2. Correlation between the skeletal and soft tissue involvement Table 3 and Fig. 5 describe this analysis and show a severe deficiency of the zygomatic bone (from 60% to almost 100% volume loss compared to the controls), a smaller orbital involvement (0e 15%), and a variable fat compartments involvement (from 20% to 45%), with the BFP being less involved than the PZF. By comparing two individuals, our goal was not to highlight statistical correlations but to show a trend indicating that the soft tissue and skeletal involvements were not always proportional. 4. Discussion Although the skeletal reconstruction provides in theory a correct projection of the orbits and zygomas, TCS patients continue to have a soft tissue deficit affecting primarily the jugal, zygomatic, orbital, and infraorbital regions. Depending on the severity, various
Fig. 4. Schematic representation of the deficit gradient (red to blue) of the superficial fat compartments from the orbit to the cheek region.
soft tissue surgical techniques are available, but they must be selected in line with the skeletal reconstruction. Alloplastic implants made of DacronÒ or GoretexÒ have been used mainly in Parry-Romberg disease (Gueganton et al., 2000) with an acceptable tolerance. However, they are limited in size and difficult to adapt to the three-dimensional soft tissue loss found in TCS. Lyophilized cartilage has been successfully used to reconstruct the mandibular and zygomatic volumetric deficits present in hemifacial microsomia (Sailer, 1983; Sailer and Farmand, 1991). Injectable biomaterials also have their theoretical indications, but unfortunately have either a significant complication rate or a limited lifespan for the safest. This filling technique is therefore not advisable in the present time, even in mild forms. With progress continuing in biomaterial tolerance, we will likely reconsider this indication in the near future. As evidenced by recent publications (Clauser et al., 2011; Güven et al., 2010; Podmelle et al., 2012), numerous reconstruction techniques exist to increase the fat component in the lateral areas of the middle and lower third of the face. Commonly used methods include structural fat grafting (Clauser et al., 2011), the transfer of pedicled dermal-adipose flaps (Güven et al., 2010), or the transfer of vascularized dermal-adipose free flaps (Podmelle et al., 2012). Dermal-fat grafts have been used to fill the hypotrophy found in Parry-Romberg disease, hemifacial microsomia, or TCS (Snyder, 1956; Kobus and Wojcicki, 2006). Their indications are limited deficits of small volumes, when the risk of graft sagging is not too important. Autologous fat grafts, extensively used since the work of Coleman (Coleman, 1998) especially in the treatment of Parry-Romberg disease (Clauser et al., 2011), are used as a first-line approach in the primary treatment and in revision of TCS reconstruction techniques (Posnick and Ruiz, 2000; Kobus and Wojcicki, 2006; Zhang et al., 2009a,b). When the lipofilling technique is chosen it should be performed prior to the bony reconstruction. The advantages of this
Fig. 5. Percentage of volume deficiency compared to the controls and comparison of the skeletal and soft tissue involvement in TCS patients. PZF: Prezygomatic fat, BFP : Buccal fat pad.
C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680
prior lipofilling are twofold: the adipose-derived stem cells present within the graft will stimulate bone repair by promoting osteoblast and osteoclast function, and the adipose tissue-derived stromal vascular fraction of the graft may favour early revascularization of the bone grafts, both promoting integration while minimizing bony resorption (Nakahara et al., 2009; Scherberich et al., 2010; Lee et al., 2011). Pedicled flaps based on temporal vessels have given rise to different therapeutic approaches, using either the temporal muscle (van der Meulen et al., 1984) or a superficial temporal fascia flap, sandwiched between one or two dermal-fat flaps (Zhang et al., 2009a,b). Microsurgical flaps also have a place in this type of surgery. Developed since the 1970’s by Antia, Tweed et al. (1984) and Longaker and Siebert (1995), the inguinal dermal-fat flaps have many disadvantages: difficult anastomosis, difficult unfolding of the digitations to adapt to the defect and a significant risk of sagging. The majority of flaps described in the literature require revision up to 2e3 times, mainly for debulking, which need to be done in a semi-sitting position. Koshima et al. (2000) and Podmelle et al. (2012) proposed the deep inferior epigastric perforator flap (DIEP) in this indication. The parascapular flap (Gilbert and Teot, 1982) is one that seems best suited to the malformation. This flap has many properties, which make it suitable for the reconstruction of TCS hypoplasia. These properties include the possibility to vary the thickness of the flap safely (Upton et al., 1992), as well as the feasibility of multiple extensions related to the dorsal thoracic fascia (Kim et al., 1987; Siebert et al., 1996), which allow an accurate architectural variation in all directions. Finally, by limiting the adipose part of the reconstructed volume, the risk of sagging can be diminished (Siebert et al., 1997). Although experienced teams show good results with this technique (Longaker and Siebert, 1995; Siebert et al., 1996; Saadeh et al., 2006), it is only useful in cases with severe soft tissue hypoplasia. The recipient vessels of choice are the superficial temporal vessels, which are accessed using an extensive facelift approach in the temporal region. This approach will allow the creation of a dissection plane in front of the superficial muscular aponeurotic system (SMAS), where the flap will lie attached to periosteal and fascial fixation points. Despite this, it often seems necessary to reoperate to resuspend the flap or reduce its volume locally. The microsurgical procedure should be planned after growth, in one or two steps. If the patient also requires an auricular reconstruction, the Brent (1980a; 1980b) method of reconstruction should used, rather than Nagata’s (1994a; 1994b; 1994c; 1994d) or Firmin’s technique (1998; 2001), to provide retroauricular coverage without using a temporoparietal fascia flap which is based on the superficial temporal artery. If Nagata’s or Firmin’s technique is chosen, the ear reconstruction should be done first. The free flap can then be anastomosed after locating the path of the plicated superficial temporal artery. Finally, microsurgical composite flaps can theoretically allow a “one-step” soft tissue and skeletal reconstruction, although no description applied to TCS can be found in the literature. The iliac crest free flap may provide up to 12 cm of bone (O’Brien et al., 1979; Stock et al., 1991; Forrest et al., 1992), which is more than enough to recreate the orbitozygomatic skeletal framework. Unfortunately, its donor site morbidity remains high, and its indication in TCS must remain a rescue solution, for instance in extremely hypotrophic cases or when all other treatment options have been ruled out. The composite parascapular flap allows the harvest of the serratus anterior muscle (Deraemaecker et al., 1988), and 15 cm of corticocancellous bone from the free edge of the scapula (Coleman and Sultan 1991). According to Swartz et al. (Swartz et al., 1986), up to three osteotomies can also be performed. This versatility makes it
679
the flap of choice in TCS. Upton et al. (Upton et al., 1992) have also reported its use in the treatment of hemifacial microsomia. 5. Conclusion The surgical management of TCS patients is a remarkable example in the treatment of craniofacial malformations. With a multidisciplinary approach, the surgeon has to integrate all components of the disease in order to intervene in a timely manner, taking into account the growth and demands of his patient and his family. In order not to repeat unnecessary or harmful surgery, the surgeon must perform the reconstruction with a minimal number of procedures by adapting the surgical protocol to the severity of the malformation. The morphometric analysis allowed us to establish an actual mapping of the tissue deficits present in TCS, and serve as a reconstruction guide to adapt our current surgical techniques to the malformation. Even if the zygomatico-orbital reconstruction is part of a primarily aesthetic process, it is the guarantor of social integration often hampered by deafness and severe maxillomandibular dysmorphia. Improving appearance of the eyes is often a priority for these syndromic patients; thus it must be integrated early in the therapeutic schedule. Ethics This study follows the principles of the World Medical Association Declaration of Helsinki. Funding This study has not been supported in the form of grants. Conflict of interest statement None. Acknowledgements This work has been made possible due to the kindliness and goodwill of the French Treacher Collins (Coline) association. References Aiache AE, Ramirez OH: The suborbicularis oculi fat pads: an anatomic and clinical study. Plast Reconstr Surg 95: 37e42, 1995 Brent B: The correction of microtia with autogenous cartilage grafts: I. The classic deformity. Plast Reconstr Surg 66: 1e12, 1980a Brent B: The correction of microtia with autogenous cartilage grafts: II. Atypical and complex deformities. Plast Reconstr Surg 66: 13e21, 1980b Clauser LC, Tieghi R, Consorti G: Parry-Romberg syndrome: volumetric regeneration by structural fat grafting technique. J Craniomaxillofac Surg 22: 1695e1701, 2011 Coleman 3rd JJ, Sultan MR: The bipedicled osteocutaneous scapula flap: a new subscapular system free flap. Plast Reconstr Surg 87: 682e692, 1991 Coleman SR: Structural fat grafting. Aesthet Surg J 18: 386e388, 1998 Deraemaecker R, Thienen CV, Lejour M, Dor R: The serratus anterior scapular free flap: a new osteomuscular unit for reconstruction after radical head and neck surgery. Proceedings of the Second International Conference on head and neck cancer, 1988 Firmin F: Ear reconstruction in cases of typical microtia. Personal experience based on 352 microtic ear corrections. Scand J Plast Reconstr Surg Hand Surg 32: 35e47, 1998 Firmin F: Auricular reconstruction in cases of microtia. Principles, methods and classification. Ann Chir Plast Esthet 46: 447e466, 2001 Forrest C, Boyd B, Manktelow R, Zuker R, Bowen V: The free vascularised iliac crest tissue transfer: donor site complications associated with eighty-two cases. Br J Plast Surg 45: 89e93, 1992 Gilbert A, Teot L: The free scapular flap. Plast Reconstr Surg 69: 601e604, 1982 Gosain AK, Klein MH, Sudhakar PV, Prost RW: A volumetric analysis of soft-tissue changes in the aging midface using high-resolution MRI: implications for facial rejuvenation. Plast Reconstr Surg 115: 1143e1152, 2005 Gueganton C, Chavoin JP, Boutault F, Mouffarege R, Papillon, Costagliola M: Treatment of facial lesions in Parry-Romberg and Barraquer-Simons
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syndromes: report of 12 clinical cases. Ann Chir Plast Esthet 45: 436e451, 2000 Güven E, Kuvat SV, Aydin HU, Yazar M, Emekli U: Facial contour reconstruction with temporoparietal prelaminated dermal-adipose flaps. J Craniomaxillofac Surg 38: 374e378, 2010 Kim PS, Gottlieb JR, Harris GD, Nagle DJ, Lewis VL: The dorsal thoracic fascia: anatomic significance with clinical applications in reconstructive microsurgery. Plast Reconstr Surg 79: 72e80, 1987 Kobus K, Wojcicki P: Surgical treatment of Treacher Collins syndrome. Ann Plast Surg 56: 549e554, 2006 Koshima I, Inagawa K, Urushibara K, Ohtsuki M, Moriguchi T: Deep inferior epigastric perforator dermal-fat or adiposal flap for correction of craniofacial contour deformities. Plast Reconstr Surg 106: 10e15, 2000 Lee K, Kim H, Kim JM, Kim JR, Kim KJ, Kim YJ, et al: Systemic transplantation of human adipose-derived stem cells stimulates bone repair by promoting osteoblast and osteoclast function. J Cell Mol Med 15: 2082e2094, 2011 Longaker MT, Siebert JW: Microvascular free-flap correction of severe hemifacial atrophy. Plast Reconstr Surg 96: 800e809, 1995 Loukas M, Kapos T, Louis Jr RG, Wartman C, Jones A, Hallner B: Gross anatomical, CT and MRI analyses of the buccal fat pad with special emphasis on volumetric variations. Surg Radiol Anat 28: 254e260, 2006 Moss CJ, Mendelson BC, Taylor GI: Surgical anatomy of the ligamentous attachments in the temple and periorbital regions. Plast Reconstr Surg 105: 1475e1490, 2000 Nagata S: Modification of the stages in total reconstruction of the auricle: part I. Grafting the three-dimensional costal cartilage framework for lobule-type microtia. Plast Reconstr Surg 93: 221e230, 1994a Nagata S: Modification of the stages in total reconstruction of the auricle: part II. Grafting the three-dimensional costal cartilage framework for concha-type microtia. Plast Reconstr Surg 93: 231e242, 1994b Nagata S: Modification of the stages in total reconstruction of the auricle: part III. Grafting the three-dimensional costal cartilage framework for small conchatype microtia. Plast Reconstr Surg 93: 243e253, 1994c Nagata S: Modification of the stages in total reconstruction of the auricle: part IV. Ear elevation for the constructed auricle. Plast Reconstr Surg 93: 254e266, 1994d Nakahara H, Misawa H, Hayashi T, Kondo E, Yuasa T, Kubota Y, et al: Bone repair by transplantation of hTERT-immortalized human mesenchymal stem cells in mice. Transplantation 88: 346e353, 2009 O’Brien BM, Morrison WA, MacLeod AM, Dooley BJ: Microvascular osteocutaneous transfer using the groin flap and iliac crest and the dorsalis pedis flap and second metatarsal. Br J Plast Surg 32: 188e206, 1979
Podmelle F, Metelmann HR, Waite P: Endoscopic abdominoplasty providing a perforator fat flap for treatment of hemi-facial microsomia. J Craniomaxillofac Surg 40: 665e667, 2012 Posnick JC, Ruiz RL: Treacher Collins syndrome: current evaluation, treatment, and future directions. Cleft Palate Craniofac J 37: 434, 2000 Rohrich RJ, Pessa JE: The fat compartments of the face: anatomy and clinical implications for cosmetic surgery. Plast Reconstr Surg 119: 2219e2227, 2007 Saadeh P, Reavey PL, Siebert JW: A soft-tissue approach to midfacial hypoplasia associated with Treacher Collins syndrome. Ann Plast Surg 56: 522e525, 2006 Sailer HF, Farmand Z: Treatment of otomandibular dysostosis. In: Pfeifer G (ed.), Craniofacial abnormalities and clefts of the lip, alveolus and palate. New York: Thieme, 102e109, 1991 Sailer HF: Transplantation of lyophilised cartilage in maxillofacial surgery, experimental foundations and clinical success. Basel: Karger, 1983 Scherberich A, Muller AM, Schafer DJ, Banfi A, Martin I: Adipose tissue-derived progenitors for engineering osteogenic and vasculogenic grafts. J Cell Physiol 225: 348e353, 2010 Siebert JW, Anson G, Longaker MT: Microsurgical correction of facial asymmetry in 60 consecutive cases. Plast Reconstr Surg 97: 354e363, 1996 Siebert JW, Longaker MT, Angrigiani C: The inframammary extended circumflex scapular flap: an aesthetic improvement of the parascapular flap. Plast Reconstr Surg 99: 70e77, 1997 Snyder CC: Bilateral facial agenesia; Treacher Collins syndrome. Am J Surg 92: 81e87,1956 Stock W, Hierner R, Dielert E, Stotz R, Manninger J, Wolf K: The iliac crest region: donor site for vascularized bone periosteal and soft tissue flaps. Ann Plast Surg 26: 105e109, 1991 Swartz WM, Banis JC, Newton ED, Ramasastry SS, Jones NF, Acland R: The osteocutaneous scapular flap for mandibular and maxillary reconstruction. Plast Reconstr Surg 77: 530e545, 1986 Tweed AE, Manktelow RT, Zuker RM: Facial contour reconstruction with free flaps. Ann Plast Surg 12: 313e320, 1984 Upton J, Albin RE, Mulliken JB, Murray JE: The use of scapular and parascapular flaps for cheek reconstruction. Plast Reconstr Surg 90: 959e971, 1992 van der Meulen JC, Hauben DJ, Vaandrager JM, Birgenhager-Frenkel DH: The use of a temporal osteoperiosteal flap for the reconstruction of malar hypoplasia in Treacher Collins syndrome. Plast Reconstr Surg 74: 687e693, 1984 Zhang Y, Jin R, Shi Y, Sun B, Qian Y: Pedicled superficial temporal fascia sandwich flap for reconstruction of severe facial depression. J Craniofac Surg 20: 505e 508, 2009a Zhang Z, Niu F, Tang X, Yu B, Liu J, Gui L: Staged reconstruction for adult complete Treacher Collins syndrome. J Craniofac Surg 20: 1433e1438, 2009b
Contents lists available at SciVerse ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Computer-assisted midface reconstruction in Treacher Collins syndrome part 2: Soft tissue reconstruction Christian Herlin a, *, Jean Charles Doucet a, b, Michèle Bigorre a, Guillaume Captier a, c a
Department of Plastic and Craniofacial Pediatric Surgery (Head: Pr Guillaume Captier), Faculty of Medicine, University of Montpellier, 371 Avenue du doyen Gaston Giraud, 34295 Montpellier Cedex 5, France b Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Dalhousie University, Halifax, Nova Scotia, Canada c Laboratory of Anatomy, Faculty of Medicine, University of Montpellier, France
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 10 June 2012 Accepted 4 January 2013
Introduction: Treacher Collins syndrome (TCS) midfacial involvement associate a skeletal hypoplasia centred on the zygoma to a hypoplasia of all surrounding soft tissues layers and an inferolateral lower lid pseudocoloboma. TCS soft tissue hypoplasia, which has not been well studied, continues to bring challenges in both the indication of surgical treatment and the prediction of their results. Material and method: From a standard magnetic resonance imaging (MRI) acquisition, we studied qualitatively and quantitatively the prezygomatic fat compartments and the buccal fat pad of two individuals with TCS whose age were 10 and 14 years. In parallel, we studied 20 controls at the same age to obtain a morphometric database of reference and compare our results. TCS soft tissue involvement was correlated to the results of our prior skeletal involvement study. Results: The midfacial fat compartments in TCS are severely hypoplastic, especially in the superficial and lateral compartments of the face (all P’s < 0.001). No significant correlation existed between the soft tissue and the skeletal involvement. Conclusions: To our knowledge, this is the first published study of TCS midfacial fat compartments. Their hypoplasia is an important part of the syndrome’s facial deformity. The knowledge of their anatomy, organization and volumetric variation is essential. Their re-establishment is key in the early treatment phases of this syndrome. Using the preoperative data, the morphometric database of reference, and surgical simulation, an appropriate surgical technique, going from an autologous fat graft to a free flap, can then be chose. Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Treacher Collins syndrome Franceschetti syndrome Soft tissue Lipofilling Fat Computer-assisted surgery Fat grafting Presurgical simulation
1. Introduction Treacher Collins syndrome (TCS) is a rare and severe craniofacial malformation with bilateral midfacial zygomatico-orbital bony involvement. The skeletal hypoplasia is centred on the zygoma, and is associated with hypoplasia of all the surrounding soft tissues layers, and an inferolateral lower lid pseudocoloboma. This soft tissue involvement presents an important aesthetic and reconstructive challenge, but remains poorly studied. The purpose of our study was to evaluate the facial fat compartments in TCS, using magnetic resonance imaging (MRI). Our specific aim was to evaluate which compartments are most affected, and in what proportion, by comparing them to a control population. This will help to guide the reconstruction and to select * Corresponding author. Tel.: þ33 (0)619715557; fax: þ33 (0)467603639. E-mail address: [email protected] (C. Herlin).
between different surgical techniques including alloplastic implants, dermal-fat graft, free flaps and autologous fat graft. 2. Materials and methods In parallel with our skeletal study, an MRI study was performed in two TCS patients aged 10 and 14 years. These patients were also evaluated with computed tomography (CT) to correlate the soft tissue and the skeletal involvement. To draw conclusions on the data acquired from the MRI and produce a morphometric database, 10 controls per age category were studied. The data studied came from 20 patients (40 “healthy” control sides) who had a craniofacial MRI for problems not related to the facial soft tissues. This database was also compared to the rare data found in the literature (Gosain et al., 2005; Loukas et al., 2006). A single examiner performed the data collection and analysis. All the MRI’s were acquired by the same machine (Philips Brilliance,
1010-5182/$ e see front matter Ó 2013 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jcms.2013.01.008
C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680
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Table 1 Buccal fat pad volumes (cm3) in TCS patients and in 10 controls per age category.
Fig. 1. Example of fatty ROI segmentation on MRI. Each ROI are represented by a colour (Buccal fat pads in yellow and green; Prezygomatic fat compartments in pink and orange).
1.5T). The smallest possible field of view was chosen to increase special resolution. T1 sequencing was performed in the axial, coronal and sagittal plane. T2 sequencing was only performed in the axial plane. Since the fat compartments were our regions of interest (ROI), no other sequences were chosen. Slice thickness was set at 1.5 mm to avoid a “dead space” effect while ensuring adequate resolution for the volumetric reconstruction. To study all prezygomatic fat compartments, the slices were realigned along a plane extending from the lateral canthus to the oral commissure, as advocated by Gosain et al., (2005). Post-image processing was realized with the MYRIANÒ expert v. 1.5.2 software (Intrasense SAS, Montpellier, France). The first step of soft tissue segmentation (Fig. 1) was carried out automatically. After manual verification, following the anatomical fat compartments description of Rohrich and Pessa (2007) and Aiache and Ramirez (1995), the following geometric parameters were determined: the buccal fat pad (BFP) volume, and the superficial and
Ages
10
Sides
R
L
R
14 L
Treacher Collins Control 1 Control 2 Control 3 Control 4 Control 5 Control 6 Control 7 Control 8 Control 9 Control 10
4.38 6.217 5.788 7.26 6.356 5.661 6.124 5.741 6.143 7.223 6.123
4.355 7.115 6.019 6.983 6.424 8.826 6.342 5.12 6.23 7.124 6.294
6.96 10.9 9.887 7.436 10.3 8.672 9.214 8.876 8.812 8.945 8.786
6.859 11 9.873 7.667 10.105 7.923 9.245 8.245 8.123 8.751 8.642
deep prezygomatic fat volumes (PZF). The prezygomatic fat volumes included the medial, middle and lateral temporal cheek fat compartments, the suborbicularis compartment, and the retroorbicularis fat compartment (Rohrich and Pessa, 2007). The volume measurement of each individual prezygomatic compartment was not possible due to the MRI resolution. The soft tissue deficiency was then compared to the skeletal deficiency found on CT, to calculate a volumetric ratio (bone vs fat) (Fig. 2). All statistics were computed with the SPSS Statistics 17.0 software (version 17.0.0, SPSS Inc, Chicago, IL). Appropriate bivariate and multivariate statistics were performed, and the level of statistical significance was set at P < 0.05. 3. Results 3.1. Fat compartments study The TCS buccal fat pad volumes found on MRI were significantly lower than the volumes found in the control population (P < 0.001) (Table 1 and Fig. 2). The control population measurements were similar to the literature data (4e6). In TCS the superficial and deep prezygomatic fat volumes were also significantly lower than the control population volumes (P < 0.001) (Table 2 and Fig. 3). The superficial fat compartments were proportionally more affected towards the orbital rim. The deficit gradient of the superficial fat compartments is shown schematically in Fig. 4. In the facial “cleft” area of TCS patients the deep fat compartments persisted. When focussing on the BFP, the temporal and masseteric extensions were more affected. Subjectively, the compartmentalization of the PZF was also increased and the fibrous tract defined as soft tissue ligaments (Moss et al., 2000; Rohrich and Pessa, 2007), which are sometimes visible on MRI, seemed more numerous and less structured in the syndromic patients.
Table 2 Prezygomatic fat volumes (cm3) in TCS patients and in 10 controls per age category.
Fig. 2. Buccal fat pad volumes (cm3) in TCS patients and in 10 controls per age category.
Ages
10
Sides
R
L
14 R
L
Treacher Collins Control 1 Control 2 Control 3 Control 4 Control 5 Control 6 Control 7 Control 8 Control 9 Control 10
7.267 13.9 12.4 13.3 14.0 12.5 14.7 13.1 13.3 12.9 13.8
7.252 14.5 13.2 12.2 13.6 12.9 14.3 12.9 13.1 12.7 13.5
10.8 19.0 22.3 17.7 20 14.9 18.1 22.1 17.3 20.2 20.8
11.4 19.3 21.9 18.1 19.7 15.3 17.6 21.6 17.1 20.4 21.1
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C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680 Table 3 Percentage of volume deficiency compared to the controls and comparison of the skeletal and soft tissue involvement in TCS patients.
Orbit Zygoma PZF BFP
Patient 1
Patient 2
Patient 3
Patient 4
3.6% 62.1% 45.6% 31.9%
4.9% 81.2% 42.3% 20.8%
15% 97.9% X X
0% 98.9% X X
PZF: Prezygomatic fat; BFP: Buccal fat pad.
Fig. 3. Prezygomatic fat volumes (cm3) in TCS patients and in 10 controls per age category.
3.2. Correlation between the skeletal and soft tissue involvement Table 3 and Fig. 5 describe this analysis and show a severe deficiency of the zygomatic bone (from 60% to almost 100% volume loss compared to the controls), a smaller orbital involvement (0e 15%), and a variable fat compartments involvement (from 20% to 45%), with the BFP being less involved than the PZF. By comparing two individuals, our goal was not to highlight statistical correlations but to show a trend indicating that the soft tissue and skeletal involvements were not always proportional. 4. Discussion Although the skeletal reconstruction provides in theory a correct projection of the orbits and zygomas, TCS patients continue to have a soft tissue deficit affecting primarily the jugal, zygomatic, orbital, and infraorbital regions. Depending on the severity, various
Fig. 4. Schematic representation of the deficit gradient (red to blue) of the superficial fat compartments from the orbit to the cheek region.
soft tissue surgical techniques are available, but they must be selected in line with the skeletal reconstruction. Alloplastic implants made of DacronÒ or GoretexÒ have been used mainly in Parry-Romberg disease (Gueganton et al., 2000) with an acceptable tolerance. However, they are limited in size and difficult to adapt to the three-dimensional soft tissue loss found in TCS. Lyophilized cartilage has been successfully used to reconstruct the mandibular and zygomatic volumetric deficits present in hemifacial microsomia (Sailer, 1983; Sailer and Farmand, 1991). Injectable biomaterials also have their theoretical indications, but unfortunately have either a significant complication rate or a limited lifespan for the safest. This filling technique is therefore not advisable in the present time, even in mild forms. With progress continuing in biomaterial tolerance, we will likely reconsider this indication in the near future. As evidenced by recent publications (Clauser et al., 2011; Güven et al., 2010; Podmelle et al., 2012), numerous reconstruction techniques exist to increase the fat component in the lateral areas of the middle and lower third of the face. Commonly used methods include structural fat grafting (Clauser et al., 2011), the transfer of pedicled dermal-adipose flaps (Güven et al., 2010), or the transfer of vascularized dermal-adipose free flaps (Podmelle et al., 2012). Dermal-fat grafts have been used to fill the hypotrophy found in Parry-Romberg disease, hemifacial microsomia, or TCS (Snyder, 1956; Kobus and Wojcicki, 2006). Their indications are limited deficits of small volumes, when the risk of graft sagging is not too important. Autologous fat grafts, extensively used since the work of Coleman (Coleman, 1998) especially in the treatment of Parry-Romberg disease (Clauser et al., 2011), are used as a first-line approach in the primary treatment and in revision of TCS reconstruction techniques (Posnick and Ruiz, 2000; Kobus and Wojcicki, 2006; Zhang et al., 2009a,b). When the lipofilling technique is chosen it should be performed prior to the bony reconstruction. The advantages of this
Fig. 5. Percentage of volume deficiency compared to the controls and comparison of the skeletal and soft tissue involvement in TCS patients. PZF: Prezygomatic fat, BFP : Buccal fat pad.
C. Herlin et al. / Journal of Cranio-Maxillo-Facial Surgery 41 (2013) 676e680
prior lipofilling are twofold: the adipose-derived stem cells present within the graft will stimulate bone repair by promoting osteoblast and osteoclast function, and the adipose tissue-derived stromal vascular fraction of the graft may favour early revascularization of the bone grafts, both promoting integration while minimizing bony resorption (Nakahara et al., 2009; Scherberich et al., 2010; Lee et al., 2011). Pedicled flaps based on temporal vessels have given rise to different therapeutic approaches, using either the temporal muscle (van der Meulen et al., 1984) or a superficial temporal fascia flap, sandwiched between one or two dermal-fat flaps (Zhang et al., 2009a,b). Microsurgical flaps also have a place in this type of surgery. Developed since the 1970’s by Antia, Tweed et al. (1984) and Longaker and Siebert (1995), the inguinal dermal-fat flaps have many disadvantages: difficult anastomosis, difficult unfolding of the digitations to adapt to the defect and a significant risk of sagging. The majority of flaps described in the literature require revision up to 2e3 times, mainly for debulking, which need to be done in a semi-sitting position. Koshima et al. (2000) and Podmelle et al. (2012) proposed the deep inferior epigastric perforator flap (DIEP) in this indication. The parascapular flap (Gilbert and Teot, 1982) is one that seems best suited to the malformation. This flap has many properties, which make it suitable for the reconstruction of TCS hypoplasia. These properties include the possibility to vary the thickness of the flap safely (Upton et al., 1992), as well as the feasibility of multiple extensions related to the dorsal thoracic fascia (Kim et al., 1987; Siebert et al., 1996), which allow an accurate architectural variation in all directions. Finally, by limiting the adipose part of the reconstructed volume, the risk of sagging can be diminished (Siebert et al., 1997). Although experienced teams show good results with this technique (Longaker and Siebert, 1995; Siebert et al., 1996; Saadeh et al., 2006), it is only useful in cases with severe soft tissue hypoplasia. The recipient vessels of choice are the superficial temporal vessels, which are accessed using an extensive facelift approach in the temporal region. This approach will allow the creation of a dissection plane in front of the superficial muscular aponeurotic system (SMAS), where the flap will lie attached to periosteal and fascial fixation points. Despite this, it often seems necessary to reoperate to resuspend the flap or reduce its volume locally. The microsurgical procedure should be planned after growth, in one or two steps. If the patient also requires an auricular reconstruction, the Brent (1980a; 1980b) method of reconstruction should used, rather than Nagata’s (1994a; 1994b; 1994c; 1994d) or Firmin’s technique (1998; 2001), to provide retroauricular coverage without using a temporoparietal fascia flap which is based on the superficial temporal artery. If Nagata’s or Firmin’s technique is chosen, the ear reconstruction should be done first. The free flap can then be anastomosed after locating the path of the plicated superficial temporal artery. Finally, microsurgical composite flaps can theoretically allow a “one-step” soft tissue and skeletal reconstruction, although no description applied to TCS can be found in the literature. The iliac crest free flap may provide up to 12 cm of bone (O’Brien et al., 1979; Stock et al., 1991; Forrest et al., 1992), which is more than enough to recreate the orbitozygomatic skeletal framework. Unfortunately, its donor site morbidity remains high, and its indication in TCS must remain a rescue solution, for instance in extremely hypotrophic cases or when all other treatment options have been ruled out. The composite parascapular flap allows the harvest of the serratus anterior muscle (Deraemaecker et al., 1988), and 15 cm of corticocancellous bone from the free edge of the scapula (Coleman and Sultan 1991). According to Swartz et al. (Swartz et al., 1986), up to three osteotomies can also be performed. This versatility makes it
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the flap of choice in TCS. Upton et al. (Upton et al., 1992) have also reported its use in the treatment of hemifacial microsomia. 5. Conclusion The surgical management of TCS patients is a remarkable example in the treatment of craniofacial malformations. With a multidisciplinary approach, the surgeon has to integrate all components of the disease in order to intervene in a timely manner, taking into account the growth and demands of his patient and his family. In order not to repeat unnecessary or harmful surgery, the surgeon must perform the reconstruction with a minimal number of procedures by adapting the surgical protocol to the severity of the malformation. The morphometric analysis allowed us to establish an actual mapping of the tissue deficits present in TCS, and serve as a reconstruction guide to adapt our current surgical techniques to the malformation. Even if the zygomatico-orbital reconstruction is part of a primarily aesthetic process, it is the guarantor of social integration often hampered by deafness and severe maxillomandibular dysmorphia. Improving appearance of the eyes is often a priority for these syndromic patients; thus it must be integrated early in the therapeutic schedule. Ethics This study follows the principles of the World Medical Association Declaration of Helsinki. Funding This study has not been supported in the form of grants. Conflict of interest statement None. Acknowledgements This work has been made possible due to the kindliness and goodwill of the French Treacher Collins (Coline) association. References Aiache AE, Ramirez OH: The suborbicularis oculi fat pads: an anatomic and clinical study. Plast Reconstr Surg 95: 37e42, 1995 Brent B: The correction of microtia with autogenous cartilage grafts: I. The classic deformity. Plast Reconstr Surg 66: 1e12, 1980a Brent B: The correction of microtia with autogenous cartilage grafts: II. Atypical and complex deformities. Plast Reconstr Surg 66: 13e21, 1980b Clauser LC, Tieghi R, Consorti G: Parry-Romberg syndrome: volumetric regeneration by structural fat grafting technique. J Craniomaxillofac Surg 22: 1695e1701, 2011 Coleman 3rd JJ, Sultan MR: The bipedicled osteocutaneous scapula flap: a new subscapular system free flap. Plast Reconstr Surg 87: 682e692, 1991 Coleman SR: Structural fat grafting. Aesthet Surg J 18: 386e388, 1998 Deraemaecker R, Thienen CV, Lejour M, Dor R: The serratus anterior scapular free flap: a new osteomuscular unit for reconstruction after radical head and neck surgery. Proceedings of the Second International Conference on head and neck cancer, 1988 Firmin F: Ear reconstruction in cases of typical microtia. Personal experience based on 352 microtic ear corrections. Scand J Plast Reconstr Surg Hand Surg 32: 35e47, 1998 Firmin F: Auricular reconstruction in cases of microtia. Principles, methods and classification. Ann Chir Plast Esthet 46: 447e466, 2001 Forrest C, Boyd B, Manktelow R, Zuker R, Bowen V: The free vascularised iliac crest tissue transfer: donor site complications associated with eighty-two cases. Br J Plast Surg 45: 89e93, 1992 Gilbert A, Teot L: The free scapular flap. Plast Reconstr Surg 69: 601e604, 1982 Gosain AK, Klein MH, Sudhakar PV, Prost RW: A volumetric analysis of soft-tissue changes in the aging midface using high-resolution MRI: implications for facial rejuvenation. Plast Reconstr Surg 115: 1143e1152, 2005 Gueganton C, Chavoin JP, Boutault F, Mouffarege R, Papillon, Costagliola M: Treatment of facial lesions in Parry-Romberg and Barraquer-Simons
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