The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
implant; and hypersensitivity reactions can be seen after the gold weight implantation procedure. After these complications, 8% to 14% of the implants are required to be revised and 2% to 7% are required to be removed.10,11 Several modifications have been tried to avoid the complications such as changing the shape and the composition of the implant, the method of fixation of the implant, and the incision site in the eyelid or covering or wrapping of the implant. Although other substances such as stainless steel, hyaluronic acid gel, and autologous cartilage graft have been used for loading the upper eyelid, platinum implant is the major opponent of the gold implant. Although platinum has higher density, biocompatibility, and flexibility than gold implants, studies have shown no significant superiority of platinum over the gold implants.12–14 Besides, platinum implants increase the cost of the procedure. Historically, in the beginning, the implants had no fixation holes and they were inserted through lateral incisions over the tars blindly. However, today, implantation through a tarsal crease incision and placement of pretarsal fixation sutures are known as the standard procedure. Additional fixation by levator aponeurosis sutures can be performed for implant fixation to avoid implant displacement.15 Choi et al16 preferred elliptic implants instead of conventional rectangular ones. They stated that these elliptic-shaped, broader, and thinner implants were better suited to the natural curvature of the eyelid, and implant extrusion or migration was not seen in a 24patient series. Coverage of the implant with fascia lata to prevent complications in gold implant procedures was performed by Egemen et al.17 They suggested coverage of the anterior aspect of the implant with fascia lata graft. In our case, the tarsal plate also lost its mass and tissue quality; therefore, we need a support layer on its posterior aspect. Thus, the implant was covered with a highquality tissue on both its anterior and posterior surface. If more severe tarsal atrophy was detected, cartilage grafts may also be required to support the tarsal plate. Foster et al7 used processed human pericardium as implant barrier in revision procedures, and they stated that they achieved acceptable coverage in most of the patients. Although experimental methods such as covering the implant with bioengineered cartilage18 and polytetraflouroethylene19 are mentioned in the literature, these methods are not available for clinical use.
CONCLUSIONS Wrapping of the implant with a tensor fascia lata sandwich graft is an effective and safe method for treatment of the migration and extrusion of the gold weight implant. By this technique, facial barrier not only covers the anterior aspect of the implant but also supports the atrophic tarsal plate. It was observed that the bulky appearance surrounding the implant diminished during a period of 6 months, and the procedure resulted in high patient satisfaction.
REFERENCES 1. Yu Y, Sun J, Chen L, et al. Lid loading for treatment of paralytic lagophthalmos. Aesthetic Plast Surg 2011;35:1165–1171 2. Pickford MA, Scamp T, Harrison DH. Morbidity after gold weight insertion into the upper lid in facial palsy. Br J Plast Surg 1992;45:460–464 3. Harrisberg BP, Singh RP, Croxson GR, et al. Long-term outcome of gold weights in patients with facial nerve palsy. Otol Neurotol 2001;22:397–400 4. Kelly SA, Sharpe DT. Gold eyelid weights in patients with facial palsy: a patient review. Plast Reconstr Surg 1992;89:436–440 5. Bair RL, Harris GJ, Lyon DV, et al. Noninfectious inflammatory response to gold weight eyelid implants. Ophthal Plast Reconstr Surg 1995;11:209–214
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6. Bladen JC, Norris JH, Malhotra R. Indications and outcomes for revision of gold weight implants in upper eyelid loading. Br J Ophthalmol 2012;96:485–489 7. Foster JA, Perry JD, Cahill KV, et al. Processed human pericardium barrier for gold weight implantation. Ophthal Plast Reconstr Surg 2004;20:107–109 8. Levine RE, Shapiro JP. Reanimation of the paralyzed eyelid with the enhanced palpebral spring or the gold weight: modern replacements for tarsorrhaphy. Facial Plast Surg 2000;16:325–336 9. Iñigo F, Chapa P, Jimenez Y, et al. Surgical treatment of lagophthalmos in facial palsy: ear cartilage graft for elongating the levator palpebrae muscle. Br J Plast Surg 1996;49:452–456 10. Manktelow RT. Use of the gold weight for lagophthalamos. Oper Tech Plast Reconstr Surg 1999;6:157 11. Rofagha S, Seiff SR. Long term results for the use of gold eyelid load weights in the management of facial palsy. Plast Reconstr Surg 2010;125:142–149 12. Silver AL, Lindsay RW, Cheney ML, et al. Thin-profile platinum eyelid weighting: a superior option in the paralyzed eye. Plast Reconstr Surg 2009;123:1697–1703 13. Bladen JC, Norris JH, Malhotra R. Cosmetic comparison of gold weight and platinum chain insertion in primary upper eyelid loading for lagophthalmos.Ophthal Plast Reconstr Surg 2012;28:171–175 14. Schrom T, Loch A, Holzl M, et al. Evaluation of a new lid implant for rehabilitation of the paralyzed eye: results of a questionnaire. Laryngorhinootologie 2006;85:38–42 15. Tower RN, Dailey RA. Gold weight implantation: a better way? Ophthal Plast Reconstr Surg 2004;20:202–206 16. Choi HY, Hong SE, Lew JM. Long-term comparison of a newly designed gold implant with the conventional implant in facial nerve paralysis. Plast Reconstr Surg 1999;104:1624–1634 17. Egemen O, Ozkaya O, Uscetin I, et al. Covering the gold weight with fascialata graft in paralytic lagophthalmos patients. Br J Oral Maxillofac Surg 2012;50:369–372 18. Monroy A, Kojima K, Ghanem MA, et al. Tissue engineered cartilage “bioshell” protective layer for subcutaneous implants. Int J Pediatr Otorhinolaryngol 2007;71:547–552 19. Jacob JT, Pendleton K, Broussard E, et al. Porous alloplastic material encasement of gold weights for the treatment of paralytic lagophthalmos. Ophthal Plast Reconstr Surg 1999;15:401–406
Chondroid Syringoma of the Nasal Dorsum Hwan Jun Choi, MD, PhD,* Eun Taik Son, MD,* Hyun Ju Lee, MD, PhD† Abstract: Chondroid syringoma (CS) is an uncommon cutaneous tumor in the head and neck, with reported incidence rate from 0.01% to 0.1%. The CS of skin is a rare type of soft tissue tumor originating from the sweat glands. We report a documented case From the Departments of *Plastic and Reconstructive Surgery and †Pathology, College of Medicine, Soonchunhyang University, Cheonan, Korea. Received May 11, 2014. Accepted for publication July 14, 2014. Address correspondence and reprint requests to Dr. Hwan Jun Choi, Department of Plastic and Reconstructive Surgery, Soonchunhyang University Cheonan Hospital, 31 Soonchunhyang 6gil, Dongnam-gu, Cheonan-si, Chuncheongnam-do 330-721, Republic of Korea; E-mail: [email protected] This work was supported by the Soonchunhyang University Research Fund. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001222
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
of CS occurring in the nasal dorsum in a 58-year-old man, which developed during the course of 1 year. The clinical, gross pathologic, and histologic findings of the tumor are described. The lesion was totally excised via transcutaneous approach and showed no evidence of recurrence after excision. Key Words: Chondroid syringoma, pleomorphic adenoma, mass, nose
C
hondroid syringoma(CS) is a skin appendage tumor histopathologically similar to the mixed tumor (pleomorphic adenoma [PA]) of the salivary glands. The CS is a mixed tumor that is composed of epithelial and mesenchymal components. It may arise in any location, including the oral cavity, pharynx, larynx, esophagus, lacrimal gland, trachea, and mediastinum, where the minor salivary glands are scattered.1 It is the most common type of salivary gland tumor and the most common tumor of the parotid gland. However, CSs of nonsalivary glands are very rare except after metastasis.1 Their usual location in the nasal region is the nasal cavity, and approximately 90% of such tumors develop from the nasal septum.2 Under a clinical diagnosis of an epidermal inclusion cyst, we performed a total excision of the tumor with the skin and with direct closure. No recurrence was found during the 1 year of postoperative follow-up. Therefore, we report here a CS exceptionally located in a dorsum of the nose.
CLINICAL REPORT A 58-year-old man presented with a 2-month history of increasing pain and swelling in the nasal dorsum. Approximately 1 year before the onset of symptoms, the patient noticed a mass in the nose that had been slowly enlarging. The patient had hypertension, which was under medical control, and no other systemic disease existed. The mass had enlarged slowly after its discovery, especially in the previous year. The patient complained of discomfort because of the size of the mass and esthetic concern. Physical examination revealed that there was approximately 0.6-cm movable soft mass with elevation of the normal-appearing skin. There was no pain or tenderness. Upon physical examination of the mass, there seemed to be no discolorations, erythema, or signs of inflammation (Fig. 1). Its clinical diagnosis was an epidermal inclusion cyst. The patient underwent surgical excision including the mass and the overlying skin under local anesthesia. In gross appearance, the size of the tumor was measured, which forms a 0.6 0.3-cm rubbery, firm mass with a well-demarcated capsular formation. Histopathologic examination revealed a biphasic appearance resulting from the mixture of the epithelium and stroma. Hematoxylin-eosin staining of the tumor showed a mixed tumor that was composed of the epithelial and mesenchymal cells. In addition, the tubules were lined with 2 layers of epithelial cells, and the stroma was composed of the myxoid and
FIGURE 1. A 0.6-cm subcutaneous mass in the nasal dorsum, which is characterized as a slowly growing mass.
FIGURE 2. Mixed tumor of the skin (CS). A, Circumscribed nodule within the dermis or subcutaneous fat ( 10.25). B, The epithelial component is composed of nests and cords of cuboidal or polygonal cells with marked cystic change. Well-developed chondroid stroma is found ( 100). C, The inner lining cells have abundant cytoplasm and vesicular nuclei; the outer myoepithelial cells have hyperchromatic spindled nuclei ( 400).
chondroid matrices. Immunohistochemical staining was performed to determine the origin of the tumor. It was positive for p63, cytokeratin, epithelial membrane antigen, and week Li-67, whereas it was negative for S-100, p53, and carcinoembryonic antigen (Fig. 2). The surgical specimen had tumor-free margins. No regional or distant metastasis was identified at the time of procedure (Fig. 3). There was no recurrence at 1 year of follow-up. The postoperative course was uneventful.
DISCUSSION The CS is an uncommon eccrine sweat gland tumor that may originate from both secretory and ductal elements of the sweat gland. The tumor clinically presents as a slow-growing, asymptomatic, painless, nonulcerated subcutaneous or intracutaneous nodule.1,2 The lesion commonly measures 0.5- to 3-cm diameter.3 The PA is the most common benign salivary gland tumor.1 Clinically, it presents as a firm nodular subcutaneous mass that grows slowly for several months or years. Most PAs develop in the major salivary glands such as the parotid, submandibular, and sublingual glands. They uncommonly develop in the scalp, eyelid, nose, cheek, upper lip, and external ear.2–4 A PA is a mixed tumor that is composed of epithelial and mesenchymal components.1,5 It commonly develops in the face, usually at 20 or 30 years, and clinically presents as a firm subcutaneous nodule that is stationary or grows slowly for several years.1 A PA arises most commonly in the parotid gland, followed by the submandibular glands, minor salivary glands, and ectopic sites such as the scalp, eyelid, nose, cheek, upper lip, and external ear.1–4 The origin of the heterotopic tissue is not clear.5 One theory suggests that the salivary tissue develops from remnants of epithelial elements that exist in early embryonic life, and the tumors arise in misplaced embryonic epithelial cells derived from ectoderm and carried via the nasal pits into the nasal cavities.6 Another theory postulates that the salivary tissue results from heteroplastic changes of epithelial structures normally found in the region.7 The authors thought that our case was similar to the first opinion, because its anatomic position was nasal embryonic fusion area. Histologically, CS consists of mixed epithelial and mesenchymal elements, with epithelial cells arranged in cords and forming tubules with a myoepithelial layer, set in a myxoid or chondroid stroma.8 Two histologic variants of this tumor are described: (1) the eccrine type with smaller lumens lined by a single row of
FIGURE 3. Postoperative finding reveals scar.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
cuboidal epithelial cells and (2) the apocrine variant with tubular and cystic branching lumina lined by 2 rows of epithelial cells.8 Cellular atypia, infiltrative margins, satellite tumor nodules, tumor necrosis, and involvement of deep structures are considered as signs of malignant transformation.8 However, the histologic features of malignant CS may be very similar to the benign type.9 There are reported cases of CS with a benign histology that turned out to be malignant.10 An immunohistochemical study may show focal positivity for keratin, vimentin, desmins, and S-100 protein in the stroma, but it is of no help in distinguishing between benign and malignant forms.8,11 The CS lesions are usually not clinically distinctive, and the diagnosis is made on excisional biopsy and microscopic examination.1,5 Although multiple treatment options have been proposed, including electrodessication, dermabrasion, and vaporization with argon or CO2 laser, the usual first-line treatment of the tumor is total excision.8 The CS is a benign tumor, and recurrence does not occur if it is completely excised.10 Rare cases of malignant CS have also been reported.12,13 These malignant forms occur more commonly in younger female patients and have a predilection for occurring on the trunk or extremities; tumors are often larger than 3 cm and are locally invasive, with rare metastasis to the bones and viscera.12,13 Malignant and large tumors (ie, greater than 3 cm) may require a wide excision with a minimum 1-cm margin to prevent local recurrence.11–13 Regional lymph node resection is indicated in the presence of clinically suspicious or palpable lymph node metastases.11–13 This report indicates that CS, although rare, should be considered in the differential diagnosis of facial tumors. Because CS does not exhibit remarkable clinical manifestation, microscopic diagnosis is needed to facilitate adequate management. We conclude that surgical excision with margin free from tumor is the treatment of choice for this benign lesion. Because of the chance of disease recurrence as well as of another CS or PA arising elsewhere, longterm follow-up is needed.
REFERENCES 1. Yim YM, Yoon JW, Seo JW, et al. Pleomorphic adenoma in the auricle. J Craniofac Surg 2009;20:951–952 2. Nardone M, Ferrara G, Nappi O, et al. Pleomorphic adenoma in unusual anatomic sites: case reports and review of literature. Acta Otorhinolaryngol Ital 2002;22:158–163 3. Porter N, Sandhu A, O'Connell TB, et al. Pleomorphic adenoma of the palpebral lobe of the lacrimal gland. Otolaryngol Head Neck Surg 2007;136:328–329 4. Badia L, Weir JN, Robinson AC. Heterotopic pleomorphic adenoma of the external nose. J Laryngol Otol 1996;110:376–378 5. Sung KY, Kim YH, Lee SK, et al. An unusual presentation of pleomorphic adenoma: nasal ala. J Craniofac Surg 2012;23:e641–e642 6. Matthew S, Ersner MD, Saltzman MA. Mixed tumour of the nasal septum. Report of a case. Laryngoscope 1944;54:287–296 7. Boffi A, Fridman L. Salivary heterotopic tissue. J Laryngol Otol 1973;87:905–907 8. Yavuzer R, Basterzi Y, Sari A, et al. Chondroid syringoma: a diagnosis more frequent than expected. Dermatol Surg 2003;29:179–181 9. Borman H, Özcan G. Chondroid syringoma at the fingertip: an unusual localization. Eur J Plast Surg 1998;21:311–313 10. Sungur N, Uysal A, Gümüs M, et al. An unusal chondroid syringoma. Dermatol Surg 2003;29:977–979 11. Metzler G, Schaumburg-Lever G, Hornstein O, et al. Malignant chondroid syringoma: immunohistopathology. Am J Dermatopathol 1996;18:83–89 12. Watson JA, Walker MM, Smith NP, et al. Malignant chondroid syringoma–a rare cause of secondary bone tumour. Clin Exp Dermatol 1991;16:306–307 13. Kiely JL, Dunne B, McCabe M, et al. Malignant chondroid syringoma presenting as multiple pulmonary nodules. Thorax 1997;52:395–396
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Use of the Buccal Fat Pad as Free Graft for Closure of Oronasal Fistula in a Cleft Palate Patient Carlos Henrique Bettoni Cruz de Castro, MSc,* Leandro Napier de Souza, PhD,* Marcelo Fernandes Santos Melo, DDS*† Abstract: Oronasal fistulas are frequent complications after cleft lip and palate surgery, with difficult treatment because of the presence of fibrotic and scarred tissue as well as the absence of local virgin tissue, representing a challenge in oral and maxillofacial surgery. The size of the fistula, its location, and the cause of the defect are important factors to determine the type of treatment and surgical technique. The use of pedicled buccal fat pad (BFP) for the repair of cleft palate has shown promising results, becoming a safe and effective method. On the other hand, the use of BFP as a free graft for oral defects has been rarely described in the literature. The current study is the first case report that shows the use of free graft of BFP in oronasal fistula after cleft lip and palate surgery and aimed to discuss the promising results of this surgical technique, suggesting it as a treatment option for anterior maxillary defects, when properly indicated. Key Words: Oronasal fistula, cleft lip and palate, buccal fat pad, free graft
O
ronasal fistulas are frequent complications after cleft lip and palate reconstructive surgery, representing a problem for patients and maxillofacial surgeons. The presence of fibrotic and scarred tissue as well as the absence of local virgin tissue in the surrounding of the fissure create difficulties in treating these fistulas, leading to high rates of relapse.1 The size of the fistula, its anatomic location, and the cause of the defect are important factors to determine the type of treatment and surgical technique to be used.1,2 Literature provides several ways to repair these fistulas, including techniques of suturing the edges, buccal flaps, palatal flaps, sliding flaps, nasolabial flap, tongue flap, bone grafts, and the use of buccal fat pad (BFP).3,4 The use of pedicled BFP for closure of oronasoantral communication was introduced by Egyedi2 in 1977 and has been used since then by many surgeons, with a high success rate, becoming a safe and effective method.1 The use of BFP as a free graft for oral defects has been rarely described in the literature. Neder5 in From the *Oral and Maxillofacial Surgery Service, Department of Dentistry, Pontifical Catholic University of Minas Gerais, Belo Horizonte; and †Oral and Maxillofacial Surgery Service, Department of Dentistry, Pontifical Catholic University of Rio Grande do Sul, Porto Alegre, Brazil. Received May 19, 2014. Accepted for publication July 14, 2014. Address correspondence and reprint requests to Marcelo Fernandes Santos Melo, DDS, Pontifícia Universidade Católica do, Rio Grande do Sul, Department of Dentistry, Post Graduation Program of Oral and Maxillofacial Surgery, Av. Ipiranga, 6681, P. 06–Partenon, Porto Alegre, RS, Brazil, 90619-900; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001225
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies
implant; and hypersensitivity reactions can be seen after the gold weight implantation procedure. After these complications, 8% to 14% of the implants are required to be revised and 2% to 7% are required to be removed.10,11 Several modifications have been tried to avoid the complications such as changing the shape and the composition of the implant, the method of fixation of the implant, and the incision site in the eyelid or covering or wrapping of the implant. Although other substances such as stainless steel, hyaluronic acid gel, and autologous cartilage graft have been used for loading the upper eyelid, platinum implant is the major opponent of the gold implant. Although platinum has higher density, biocompatibility, and flexibility than gold implants, studies have shown no significant superiority of platinum over the gold implants.12–14 Besides, platinum implants increase the cost of the procedure. Historically, in the beginning, the implants had no fixation holes and they were inserted through lateral incisions over the tars blindly. However, today, implantation through a tarsal crease incision and placement of pretarsal fixation sutures are known as the standard procedure. Additional fixation by levator aponeurosis sutures can be performed for implant fixation to avoid implant displacement.15 Choi et al16 preferred elliptic implants instead of conventional rectangular ones. They stated that these elliptic-shaped, broader, and thinner implants were better suited to the natural curvature of the eyelid, and implant extrusion or migration was not seen in a 24patient series. Coverage of the implant with fascia lata to prevent complications in gold implant procedures was performed by Egemen et al.17 They suggested coverage of the anterior aspect of the implant with fascia lata graft. In our case, the tarsal plate also lost its mass and tissue quality; therefore, we need a support layer on its posterior aspect. Thus, the implant was covered with a highquality tissue on both its anterior and posterior surface. If more severe tarsal atrophy was detected, cartilage grafts may also be required to support the tarsal plate. Foster et al7 used processed human pericardium as implant barrier in revision procedures, and they stated that they achieved acceptable coverage in most of the patients. Although experimental methods such as covering the implant with bioengineered cartilage18 and polytetraflouroethylene19 are mentioned in the literature, these methods are not available for clinical use.
CONCLUSIONS Wrapping of the implant with a tensor fascia lata sandwich graft is an effective and safe method for treatment of the migration and extrusion of the gold weight implant. By this technique, facial barrier not only covers the anterior aspect of the implant but also supports the atrophic tarsal plate. It was observed that the bulky appearance surrounding the implant diminished during a period of 6 months, and the procedure resulted in high patient satisfaction.
REFERENCES 1. Yu Y, Sun J, Chen L, et al. Lid loading for treatment of paralytic lagophthalmos. Aesthetic Plast Surg 2011;35:1165–1171 2. Pickford MA, Scamp T, Harrison DH. Morbidity after gold weight insertion into the upper lid in facial palsy. Br J Plast Surg 1992;45:460–464 3. Harrisberg BP, Singh RP, Croxson GR, et al. Long-term outcome of gold weights in patients with facial nerve palsy. Otol Neurotol 2001;22:397–400 4. Kelly SA, Sharpe DT. Gold eyelid weights in patients with facial palsy: a patient review. Plast Reconstr Surg 1992;89:436–440 5. Bair RL, Harris GJ, Lyon DV, et al. Noninfectious inflammatory response to gold weight eyelid implants. Ophthal Plast Reconstr Surg 1995;11:209–214
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6. Bladen JC, Norris JH, Malhotra R. Indications and outcomes for revision of gold weight implants in upper eyelid loading. Br J Ophthalmol 2012;96:485–489 7. Foster JA, Perry JD, Cahill KV, et al. Processed human pericardium barrier for gold weight implantation. Ophthal Plast Reconstr Surg 2004;20:107–109 8. Levine RE, Shapiro JP. Reanimation of the paralyzed eyelid with the enhanced palpebral spring or the gold weight: modern replacements for tarsorrhaphy. Facial Plast Surg 2000;16:325–336 9. Iñigo F, Chapa P, Jimenez Y, et al. Surgical treatment of lagophthalmos in facial palsy: ear cartilage graft for elongating the levator palpebrae muscle. Br J Plast Surg 1996;49:452–456 10. Manktelow RT. Use of the gold weight for lagophthalamos. Oper Tech Plast Reconstr Surg 1999;6:157 11. Rofagha S, Seiff SR. Long term results for the use of gold eyelid load weights in the management of facial palsy. Plast Reconstr Surg 2010;125:142–149 12. Silver AL, Lindsay RW, Cheney ML, et al. Thin-profile platinum eyelid weighting: a superior option in the paralyzed eye. Plast Reconstr Surg 2009;123:1697–1703 13. Bladen JC, Norris JH, Malhotra R. Cosmetic comparison of gold weight and platinum chain insertion in primary upper eyelid loading for lagophthalmos.Ophthal Plast Reconstr Surg 2012;28:171–175 14. Schrom T, Loch A, Holzl M, et al. Evaluation of a new lid implant for rehabilitation of the paralyzed eye: results of a questionnaire. Laryngorhinootologie 2006;85:38–42 15. Tower RN, Dailey RA. Gold weight implantation: a better way? Ophthal Plast Reconstr Surg 2004;20:202–206 16. Choi HY, Hong SE, Lew JM. Long-term comparison of a newly designed gold implant with the conventional implant in facial nerve paralysis. Plast Reconstr Surg 1999;104:1624–1634 17. Egemen O, Ozkaya O, Uscetin I, et al. Covering the gold weight with fascialata graft in paralytic lagophthalmos patients. Br J Oral Maxillofac Surg 2012;50:369–372 18. Monroy A, Kojima K, Ghanem MA, et al. Tissue engineered cartilage “bioshell” protective layer for subcutaneous implants. Int J Pediatr Otorhinolaryngol 2007;71:547–552 19. Jacob JT, Pendleton K, Broussard E, et al. Porous alloplastic material encasement of gold weights for the treatment of paralytic lagophthalmos. Ophthal Plast Reconstr Surg 1999;15:401–406
Chondroid Syringoma of the Nasal Dorsum Hwan Jun Choi, MD, PhD,* Eun Taik Son, MD,* Hyun Ju Lee, MD, PhD† Abstract: Chondroid syringoma (CS) is an uncommon cutaneous tumor in the head and neck, with reported incidence rate from 0.01% to 0.1%. The CS of skin is a rare type of soft tissue tumor originating from the sweat glands. We report a documented case From the Departments of *Plastic and Reconstructive Surgery and †Pathology, College of Medicine, Soonchunhyang University, Cheonan, Korea. Received May 11, 2014. Accepted for publication July 14, 2014. Address correspondence and reprint requests to Dr. Hwan Jun Choi, Department of Plastic and Reconstructive Surgery, Soonchunhyang University Cheonan Hospital, 31 Soonchunhyang 6gil, Dongnam-gu, Cheonan-si, Chuncheongnam-do 330-721, Republic of Korea; E-mail: [email protected] This work was supported by the Soonchunhyang University Research Fund. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001222
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
of CS occurring in the nasal dorsum in a 58-year-old man, which developed during the course of 1 year. The clinical, gross pathologic, and histologic findings of the tumor are described. The lesion was totally excised via transcutaneous approach and showed no evidence of recurrence after excision. Key Words: Chondroid syringoma, pleomorphic adenoma, mass, nose
C
hondroid syringoma(CS) is a skin appendage tumor histopathologically similar to the mixed tumor (pleomorphic adenoma [PA]) of the salivary glands. The CS is a mixed tumor that is composed of epithelial and mesenchymal components. It may arise in any location, including the oral cavity, pharynx, larynx, esophagus, lacrimal gland, trachea, and mediastinum, where the minor salivary glands are scattered.1 It is the most common type of salivary gland tumor and the most common tumor of the parotid gland. However, CSs of nonsalivary glands are very rare except after metastasis.1 Their usual location in the nasal region is the nasal cavity, and approximately 90% of such tumors develop from the nasal septum.2 Under a clinical diagnosis of an epidermal inclusion cyst, we performed a total excision of the tumor with the skin and with direct closure. No recurrence was found during the 1 year of postoperative follow-up. Therefore, we report here a CS exceptionally located in a dorsum of the nose.
CLINICAL REPORT A 58-year-old man presented with a 2-month history of increasing pain and swelling in the nasal dorsum. Approximately 1 year before the onset of symptoms, the patient noticed a mass in the nose that had been slowly enlarging. The patient had hypertension, which was under medical control, and no other systemic disease existed. The mass had enlarged slowly after its discovery, especially in the previous year. The patient complained of discomfort because of the size of the mass and esthetic concern. Physical examination revealed that there was approximately 0.6-cm movable soft mass with elevation of the normal-appearing skin. There was no pain or tenderness. Upon physical examination of the mass, there seemed to be no discolorations, erythema, or signs of inflammation (Fig. 1). Its clinical diagnosis was an epidermal inclusion cyst. The patient underwent surgical excision including the mass and the overlying skin under local anesthesia. In gross appearance, the size of the tumor was measured, which forms a 0.6 0.3-cm rubbery, firm mass with a well-demarcated capsular formation. Histopathologic examination revealed a biphasic appearance resulting from the mixture of the epithelium and stroma. Hematoxylin-eosin staining of the tumor showed a mixed tumor that was composed of the epithelial and mesenchymal cells. In addition, the tubules were lined with 2 layers of epithelial cells, and the stroma was composed of the myxoid and
FIGURE 1. A 0.6-cm subcutaneous mass in the nasal dorsum, which is characterized as a slowly growing mass.
FIGURE 2. Mixed tumor of the skin (CS). A, Circumscribed nodule within the dermis or subcutaneous fat ( 10.25). B, The epithelial component is composed of nests and cords of cuboidal or polygonal cells with marked cystic change. Well-developed chondroid stroma is found ( 100). C, The inner lining cells have abundant cytoplasm and vesicular nuclei; the outer myoepithelial cells have hyperchromatic spindled nuclei ( 400).
chondroid matrices. Immunohistochemical staining was performed to determine the origin of the tumor. It was positive for p63, cytokeratin, epithelial membrane antigen, and week Li-67, whereas it was negative for S-100, p53, and carcinoembryonic antigen (Fig. 2). The surgical specimen had tumor-free margins. No regional or distant metastasis was identified at the time of procedure (Fig. 3). There was no recurrence at 1 year of follow-up. The postoperative course was uneventful.
DISCUSSION The CS is an uncommon eccrine sweat gland tumor that may originate from both secretory and ductal elements of the sweat gland. The tumor clinically presents as a slow-growing, asymptomatic, painless, nonulcerated subcutaneous or intracutaneous nodule.1,2 The lesion commonly measures 0.5- to 3-cm diameter.3 The PA is the most common benign salivary gland tumor.1 Clinically, it presents as a firm nodular subcutaneous mass that grows slowly for several months or years. Most PAs develop in the major salivary glands such as the parotid, submandibular, and sublingual glands. They uncommonly develop in the scalp, eyelid, nose, cheek, upper lip, and external ear.2–4 A PA is a mixed tumor that is composed of epithelial and mesenchymal components.1,5 It commonly develops in the face, usually at 20 or 30 years, and clinically presents as a firm subcutaneous nodule that is stationary or grows slowly for several years.1 A PA arises most commonly in the parotid gland, followed by the submandibular glands, minor salivary glands, and ectopic sites such as the scalp, eyelid, nose, cheek, upper lip, and external ear.1–4 The origin of the heterotopic tissue is not clear.5 One theory suggests that the salivary tissue develops from remnants of epithelial elements that exist in early embryonic life, and the tumors arise in misplaced embryonic epithelial cells derived from ectoderm and carried via the nasal pits into the nasal cavities.6 Another theory postulates that the salivary tissue results from heteroplastic changes of epithelial structures normally found in the region.7 The authors thought that our case was similar to the first opinion, because its anatomic position was nasal embryonic fusion area. Histologically, CS consists of mixed epithelial and mesenchymal elements, with epithelial cells arranged in cords and forming tubules with a myoepithelial layer, set in a myxoid or chondroid stroma.8 Two histologic variants of this tumor are described: (1) the eccrine type with smaller lumens lined by a single row of
FIGURE 3. Postoperative finding reveals scar.
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 26, Number 1, January 2015
Brief Clinical Studies
cuboidal epithelial cells and (2) the apocrine variant with tubular and cystic branching lumina lined by 2 rows of epithelial cells.8 Cellular atypia, infiltrative margins, satellite tumor nodules, tumor necrosis, and involvement of deep structures are considered as signs of malignant transformation.8 However, the histologic features of malignant CS may be very similar to the benign type.9 There are reported cases of CS with a benign histology that turned out to be malignant.10 An immunohistochemical study may show focal positivity for keratin, vimentin, desmins, and S-100 protein in the stroma, but it is of no help in distinguishing between benign and malignant forms.8,11 The CS lesions are usually not clinically distinctive, and the diagnosis is made on excisional biopsy and microscopic examination.1,5 Although multiple treatment options have been proposed, including electrodessication, dermabrasion, and vaporization with argon or CO2 laser, the usual first-line treatment of the tumor is total excision.8 The CS is a benign tumor, and recurrence does not occur if it is completely excised.10 Rare cases of malignant CS have also been reported.12,13 These malignant forms occur more commonly in younger female patients and have a predilection for occurring on the trunk or extremities; tumors are often larger than 3 cm and are locally invasive, with rare metastasis to the bones and viscera.12,13 Malignant and large tumors (ie, greater than 3 cm) may require a wide excision with a minimum 1-cm margin to prevent local recurrence.11–13 Regional lymph node resection is indicated in the presence of clinically suspicious or palpable lymph node metastases.11–13 This report indicates that CS, although rare, should be considered in the differential diagnosis of facial tumors. Because CS does not exhibit remarkable clinical manifestation, microscopic diagnosis is needed to facilitate adequate management. We conclude that surgical excision with margin free from tumor is the treatment of choice for this benign lesion. Because of the chance of disease recurrence as well as of another CS or PA arising elsewhere, longterm follow-up is needed.
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Use of the Buccal Fat Pad as Free Graft for Closure of Oronasal Fistula in a Cleft Palate Patient Carlos Henrique Bettoni Cruz de Castro, MSc,* Leandro Napier de Souza, PhD,* Marcelo Fernandes Santos Melo, DDS*† Abstract: Oronasal fistulas are frequent complications after cleft lip and palate surgery, with difficult treatment because of the presence of fibrotic and scarred tissue as well as the absence of local virgin tissue, representing a challenge in oral and maxillofacial surgery. The size of the fistula, its location, and the cause of the defect are important factors to determine the type of treatment and surgical technique. The use of pedicled buccal fat pad (BFP) for the repair of cleft palate has shown promising results, becoming a safe and effective method. On the other hand, the use of BFP as a free graft for oral defects has been rarely described in the literature. The current study is the first case report that shows the use of free graft of BFP in oronasal fistula after cleft lip and palate surgery and aimed to discuss the promising results of this surgical technique, suggesting it as a treatment option for anterior maxillary defects, when properly indicated. Key Words: Oronasal fistula, cleft lip and palate, buccal fat pad, free graft
O
ronasal fistulas are frequent complications after cleft lip and palate reconstructive surgery, representing a problem for patients and maxillofacial surgeons. The presence of fibrotic and scarred tissue as well as the absence of local virgin tissue in the surrounding of the fissure create difficulties in treating these fistulas, leading to high rates of relapse.1 The size of the fistula, its anatomic location, and the cause of the defect are important factors to determine the type of treatment and surgical technique to be used.1,2 Literature provides several ways to repair these fistulas, including techniques of suturing the edges, buccal flaps, palatal flaps, sliding flaps, nasolabial flap, tongue flap, bone grafts, and the use of buccal fat pad (BFP).3,4 The use of pedicled BFP for closure of oronasoantral communication was introduced by Egyedi2 in 1977 and has been used since then by many surgeons, with a high success rate, becoming a safe and effective method.1 The use of BFP as a free graft for oral defects has been rarely described in the literature. Neder5 in From the *Oral and Maxillofacial Surgery Service, Department of Dentistry, Pontifical Catholic University of Minas Gerais, Belo Horizonte; and †Oral and Maxillofacial Surgery Service, Department of Dentistry, Pontifical Catholic University of Rio Grande do Sul, Porto Alegre, Brazil. Received May 19, 2014. Accepted for publication July 14, 2014. Address correspondence and reprint requests to Marcelo Fernandes Santos Melo, DDS, Pontifícia Universidade Católica do, Rio Grande do Sul, Department of Dentistry, Post Graduation Program of Oral and Maxillofacial Surgery, Av. Ipiranga, 6681, P. 06–Partenon, Porto Alegre, RS, Brazil, 90619-900; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001225
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
