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Endoscopic resection of large sinonasal ossifying fibroma
American Journal of Otolaryngology –Head and Neck Medicine and Surgery 26 (2005) 54 – 56
Case reports
Endoscopic resection of large sinonasal ossifying fibroma Garrett Post, MS, Stilianos E. Kountakis, MD, PhD* Department of Otolaryngology –Head and Neck Surgery, Medical College of Georgia, Augusta, GA 30912-4060 USA Received 23 January 2004
Abstract
Fibroosseous lesions of the maxilla and paranasal sinuses differ from one another in their prognosis and treatment, with the most important distinction being that of an ossifying fibroma (OF) and fibrous dysplasia. A clinically significant OF with its potentially more aggressive behavior must be completely resected. A look at historical and current approaches along with a case report of a 19year-old woman with a recurrent sinonasal OF removed using endoscopic techniques are discussed. The case adds to the growing amount of literature showing a successful alternative to open surgery for large benign sinonasal tumors, when the character of the tumor, desire of the patient, and expertise of the physician permit endoscopic resection. With the improving techniques of sinonasal endoscopy, better care can be provided with less invasive surgery resulting in less recovery time, more aesthetically pleasing results, and decreased potential for infection. D 2005 Elsevier Inc. All rights reserved.
Ossifying fibroma (OF) is a rare fibroosseous benign tumor that has shown aggressive growth in the maxilla and paranasal sinuses resulting in considerable patient morbidity. Mandibular OF is traditionally resected using curettage because of the favorable results in this anatomic location. However, recurrence with deleterious effects when OF is located in extramandibular regions was the impetus for open en bloc resection of the tumor. The advent of sinonasal endoscopy, surgical instrumentation, improving techniques, and an increased experienced physician base make endoscopic resection of large benign sinonasal tumors possible. Endoscopic techniques for benign sinonasal tumors should always be considered because of the decreased morbidity experienced by the patients and the ability for postoperative surveillance the technique provides. This case shows a successful experience of endoscopic removal of a uniquely located OF. 1. Case report A 19-year-old white woman with a history of a right intranasal OF that was partially excised via a lateral rhinotomy at the age of 9 presented with the sensation of right eye pressure and a hard fullness in the right medial canthal* Corresponding author. E-mail address: [email protected] (S.E. Kountakis). 0196-0709/$ – see front matter D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.amjoto.2004.06.009
paranasal area that protruded outward causing a visible deformity. Endoscopic examination revealed a large firm tumor filling the right nasal cavity (Fig. 1). On a coronal sinus computed tomography scan, the tumor was seen filling the right nasal cavity with erosion of the nasal bones and tumor protrusion into the area of the right medial canthus (Fig. 2A). The tumor also eroded the lamina papyracea, caused bowing of the nasal septum toward the left nasal cavity, and pushed onto the skull base tilting the cribriform plate and crista galli toward the left (Fig. 2B). The tumor was resected endoscopically using powered straight and curved microdebriders and drill bits. At the time of surgery, the tumor was found to attach to the skull base and to have caused significant erosion of the lamina papyracea. The right periorbita and skull base were not violated and orbital fat never protruded into the surgical field. Routine postoperative endoscopic debridement of the right sinonasal cavity was performed until no evidence of crusting was seen (Fig. 3). The patient did not have evidence of recurrence by nasal endoscopy 2 years after surgery.
2. Discussion OF is classified as a benign fibroosseous lesion, a term that is synonymous with a variety of other terms (Table 1) used in the current literature. The origin of OF is debated with 1 theory claiming the origin of the tumor from periodontal roots
G. Post, S.E. Kountakis / American Journal of Otolaryngology –Head and Neck Medicine and Surgery 26 (2005) 54–56
Fig. 1. Endoscopic view of large tumor (solid arrow) present in the right nasal cavity.
because of their capacity to produce cementum and osteoid characteristics. However, Marvel et al [1] state that OF originates from primitive mesenchymal cells that are believed to produce cementum at sites distant from odontogenic tissue.
55
Fig. 3. Endoscopic view of the right middle meatus 4 weeks after surgery.
Regardless of the theory of origin, fibroosseous lesions are differentiated into OF and fibrous dysplasia that vary not only in histological and radiologic presentation but also exhibit unique and distinct clinical behaviors. Common among fibroosseous lesions is the presence of calcification and ossification. The primary distinction between OF and fibrous dysplasia is the histological presence of lamellar bone and peripheral osteoblasts in an OF and the absence thereof in fibrous dysplasia. Radiologically, OF presents as an expansile lesion with sharp demarcation from adjacent bone, whereas fibrous dysplasia tends to have more diffuse margins. OFs grow at different rates and may cause mass effect with displacement of normal bone, whereas fibrous dysplasia growth is selflimiting with skeletal maturation when present in children and often cessation of growth once adulthood is reached. In adults, fibrous dysplasia can diffusely invade bone. A tumorTs identity cannot be definitively diagnosed with radiologic imaging but by histological and gross examination of the resected material. The gross presentation of an OF is dry; avascular; yellow white in color; and either crumbly, cheesy, or gritty with a rare presentation as gelatinous or cystic with clear or straw-colored fluid [2]. OFs typically present in the mandible (75%) and are often lesions seen and treated by oral surgeons [3]. Other locations in which OFs present are the maxilla, paranasal
Table 1 Various terms that are based on histological variants of ossifying fibromas
Fig. 2. (A) Coronal sinus computed tomography scan showing a large tumor with erosion of the nasal bones and tumor protrusion into the area of the right medial canthus. (B) Coronal sinus computed tomography scan showing a large tumor causing erosion of the lamina papyracea and tilting the cribriform plate and crista galli toward the left.
Ossifying fibroma Cementifying fibroma Cemento-ossifying fibroma Desmo-osteoblastoma Psammo-osteoid fibroma Psammonmatoid ossifying fibroma Juvenile ossifying fibroma Juvenile aggressive ossifying fibroma Juvenile active ossifying fibroma
56
G. Post, S.E. Kountakis / American Journal of Otolaryngology –Head and Neck Medicine and Surgery 26 (2005) 54–56
sinuses, and 3 reported cases in the temporal bone [4]. The otolaryngologistsT concern lies with the extramandibular presentation because OFs are believed to behave more aggressively than their mandibular counterparts and require surgical intervention with complete resection [3]. Presenting symptoms of OF are dependent on the location of the tumor and can range from nasal obstruction to disfiguration. Individual patients may present with ocular disturbances, such as proptosis, intracranial extension, and mucoceles. Patients with reported OF range from ages 3 months to 72 years with more aggressive cases presenting at an earlier age [2]. The popular belief is that the highest incidence occurs between the ages of 20 to 40 years with a predilection for women [1]. 3. Treatment approaches Radiotherapy is contraindicated as treatment for OFs because it may increase malignant transformation rates from 0.4% to 40% [4]. Simple curettage has been the traditional means by which to treat OF of the mandible, but the more aggressive nature and higher recurrence rates of OFs localized outside of the mandible demand wide local excision [1]. Complete resection of OF is curative, and the exact surgical approach depends on the location and extent of the tumor. The following procedures have been successfully performed and described in the literature: (1) Caldwell-Luc, curettage, with peripheral ostectomy when the tumor was located in the floor of the maxillary sinus [5]; (2) lateral rhinotomy with medial maxillectomy when the tumor was located in the medical wall of the maxillary sinus [1]; (3) total maxillectomy for recurrent maxillary tumor1; (4) external ethmoidectomy for recurrent ethmoid tumor [4]; and (5) a total 13 sinonasal cases managed endoscopically and published in the literature [3,6,7]. Endoscopic advantages include direct visualization, enhanced magnification, no external deformity, and decreased morbidity versus open surgery. Although clinically
significant OF is a rare disease, endoscopic advantages are evident from the number of other uses that have benefited from endoscopic techniques. Complications of the endoscopic technique include injury the skull base with resultant cerebrospinal fluid rhinorrhea, which can be repaired endoscopically during the same setting [7]. In this report, we describe a case of a large OF that was resected endoscopically without complications and without recurrence at the 2-year follow-up. 4. Conclusion Endoscopic resection of sinonasal ossifying fibromas is an excellent therapeutic option when done by a surgeon experienced in endoscopic sinonasal surgery. The tumorTs well-demarcated borders allow for complete resection and promote assurance of tumor-free margins, although recurrence is always possible. The additional advantage that the endoscopic provides in these cases is excellent postoperative surveillance for tumor recurrence. References [1] Marvel JB, Marsh MA, Catlin FI. Ossifying fibroma of the mid-face and paranasal sinuses: diagnostic and therapeutic considerations. Otolaryngol Head Neck Surg 1991;104:803 - 8. [2] Johnson LC, Yousefi M, Vinh TN, et al. Juvenile active ossifying fibroma: its nature, dynamics and origin. Acta Otolaryngol Suppl (Stockh) 1991;488:1 - 40. [3] London SD, Schlosser RJ, Gross CW. Endoscopic management of benign sinonasal tumors: a decade of experience. Am J Rhinol 2002; 16:221 - 7. [4] Vaidya AM, Chow JM, Goldberg K, et al. Juvenile aggressive ossifying fibroma presenting as an ethmoid sinus mucocele. Otolaryngol Head Neck Surg 1998;119:665 - 8. [5] Chong VFH, Tan LHC. Maxillary sinus ossifying fibroma. Am J Otolaryngol 1997;18:419 - 24. [6] Choi YC, Jeon EJ, Park YS. Ossifying fibroma arising in the right ethmoid sinus and nasal cavity. Int J Pediatr Otorhinolaryngol 2000; 54:159 - 62. [7] Brodish BN, Morgan CE, Sillers MJ. Endoscopic resection of fibroosseous lesions of the paranasal sinuses. Am J Rhinol 1999;13:111 - 6.
Case reports
Endoscopic resection of large sinonasal ossifying fibroma Garrett Post, MS, Stilianos E. Kountakis, MD, PhD* Department of Otolaryngology –Head and Neck Surgery, Medical College of Georgia, Augusta, GA 30912-4060 USA Received 23 January 2004
Abstract
Fibroosseous lesions of the maxilla and paranasal sinuses differ from one another in their prognosis and treatment, with the most important distinction being that of an ossifying fibroma (OF) and fibrous dysplasia. A clinically significant OF with its potentially more aggressive behavior must be completely resected. A look at historical and current approaches along with a case report of a 19year-old woman with a recurrent sinonasal OF removed using endoscopic techniques are discussed. The case adds to the growing amount of literature showing a successful alternative to open surgery for large benign sinonasal tumors, when the character of the tumor, desire of the patient, and expertise of the physician permit endoscopic resection. With the improving techniques of sinonasal endoscopy, better care can be provided with less invasive surgery resulting in less recovery time, more aesthetically pleasing results, and decreased potential for infection. D 2005 Elsevier Inc. All rights reserved.
Ossifying fibroma (OF) is a rare fibroosseous benign tumor that has shown aggressive growth in the maxilla and paranasal sinuses resulting in considerable patient morbidity. Mandibular OF is traditionally resected using curettage because of the favorable results in this anatomic location. However, recurrence with deleterious effects when OF is located in extramandibular regions was the impetus for open en bloc resection of the tumor. The advent of sinonasal endoscopy, surgical instrumentation, improving techniques, and an increased experienced physician base make endoscopic resection of large benign sinonasal tumors possible. Endoscopic techniques for benign sinonasal tumors should always be considered because of the decreased morbidity experienced by the patients and the ability for postoperative surveillance the technique provides. This case shows a successful experience of endoscopic removal of a uniquely located OF. 1. Case report A 19-year-old white woman with a history of a right intranasal OF that was partially excised via a lateral rhinotomy at the age of 9 presented with the sensation of right eye pressure and a hard fullness in the right medial canthal* Corresponding author. E-mail address: [email protected] (S.E. Kountakis). 0196-0709/$ – see front matter D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.amjoto.2004.06.009
paranasal area that protruded outward causing a visible deformity. Endoscopic examination revealed a large firm tumor filling the right nasal cavity (Fig. 1). On a coronal sinus computed tomography scan, the tumor was seen filling the right nasal cavity with erosion of the nasal bones and tumor protrusion into the area of the right medial canthus (Fig. 2A). The tumor also eroded the lamina papyracea, caused bowing of the nasal septum toward the left nasal cavity, and pushed onto the skull base tilting the cribriform plate and crista galli toward the left (Fig. 2B). The tumor was resected endoscopically using powered straight and curved microdebriders and drill bits. At the time of surgery, the tumor was found to attach to the skull base and to have caused significant erosion of the lamina papyracea. The right periorbita and skull base were not violated and orbital fat never protruded into the surgical field. Routine postoperative endoscopic debridement of the right sinonasal cavity was performed until no evidence of crusting was seen (Fig. 3). The patient did not have evidence of recurrence by nasal endoscopy 2 years after surgery.
2. Discussion OF is classified as a benign fibroosseous lesion, a term that is synonymous with a variety of other terms (Table 1) used in the current literature. The origin of OF is debated with 1 theory claiming the origin of the tumor from periodontal roots
G. Post, S.E. Kountakis / American Journal of Otolaryngology –Head and Neck Medicine and Surgery 26 (2005) 54–56
Fig. 1. Endoscopic view of large tumor (solid arrow) present in the right nasal cavity.
because of their capacity to produce cementum and osteoid characteristics. However, Marvel et al [1] state that OF originates from primitive mesenchymal cells that are believed to produce cementum at sites distant from odontogenic tissue.
55
Fig. 3. Endoscopic view of the right middle meatus 4 weeks after surgery.
Regardless of the theory of origin, fibroosseous lesions are differentiated into OF and fibrous dysplasia that vary not only in histological and radiologic presentation but also exhibit unique and distinct clinical behaviors. Common among fibroosseous lesions is the presence of calcification and ossification. The primary distinction between OF and fibrous dysplasia is the histological presence of lamellar bone and peripheral osteoblasts in an OF and the absence thereof in fibrous dysplasia. Radiologically, OF presents as an expansile lesion with sharp demarcation from adjacent bone, whereas fibrous dysplasia tends to have more diffuse margins. OFs grow at different rates and may cause mass effect with displacement of normal bone, whereas fibrous dysplasia growth is selflimiting with skeletal maturation when present in children and often cessation of growth once adulthood is reached. In adults, fibrous dysplasia can diffusely invade bone. A tumorTs identity cannot be definitively diagnosed with radiologic imaging but by histological and gross examination of the resected material. The gross presentation of an OF is dry; avascular; yellow white in color; and either crumbly, cheesy, or gritty with a rare presentation as gelatinous or cystic with clear or straw-colored fluid [2]. OFs typically present in the mandible (75%) and are often lesions seen and treated by oral surgeons [3]. Other locations in which OFs present are the maxilla, paranasal
Table 1 Various terms that are based on histological variants of ossifying fibromas
Fig. 2. (A) Coronal sinus computed tomography scan showing a large tumor with erosion of the nasal bones and tumor protrusion into the area of the right medial canthus. (B) Coronal sinus computed tomography scan showing a large tumor causing erosion of the lamina papyracea and tilting the cribriform plate and crista galli toward the left.
Ossifying fibroma Cementifying fibroma Cemento-ossifying fibroma Desmo-osteoblastoma Psammo-osteoid fibroma Psammonmatoid ossifying fibroma Juvenile ossifying fibroma Juvenile aggressive ossifying fibroma Juvenile active ossifying fibroma
56
G. Post, S.E. Kountakis / American Journal of Otolaryngology –Head and Neck Medicine and Surgery 26 (2005) 54–56
sinuses, and 3 reported cases in the temporal bone [4]. The otolaryngologistsT concern lies with the extramandibular presentation because OFs are believed to behave more aggressively than their mandibular counterparts and require surgical intervention with complete resection [3]. Presenting symptoms of OF are dependent on the location of the tumor and can range from nasal obstruction to disfiguration. Individual patients may present with ocular disturbances, such as proptosis, intracranial extension, and mucoceles. Patients with reported OF range from ages 3 months to 72 years with more aggressive cases presenting at an earlier age [2]. The popular belief is that the highest incidence occurs between the ages of 20 to 40 years with a predilection for women [1]. 3. Treatment approaches Radiotherapy is contraindicated as treatment for OFs because it may increase malignant transformation rates from 0.4% to 40% [4]. Simple curettage has been the traditional means by which to treat OF of the mandible, but the more aggressive nature and higher recurrence rates of OFs localized outside of the mandible demand wide local excision [1]. Complete resection of OF is curative, and the exact surgical approach depends on the location and extent of the tumor. The following procedures have been successfully performed and described in the literature: (1) Caldwell-Luc, curettage, with peripheral ostectomy when the tumor was located in the floor of the maxillary sinus [5]; (2) lateral rhinotomy with medial maxillectomy when the tumor was located in the medical wall of the maxillary sinus [1]; (3) total maxillectomy for recurrent maxillary tumor1; (4) external ethmoidectomy for recurrent ethmoid tumor [4]; and (5) a total 13 sinonasal cases managed endoscopically and published in the literature [3,6,7]. Endoscopic advantages include direct visualization, enhanced magnification, no external deformity, and decreased morbidity versus open surgery. Although clinically
significant OF is a rare disease, endoscopic advantages are evident from the number of other uses that have benefited from endoscopic techniques. Complications of the endoscopic technique include injury the skull base with resultant cerebrospinal fluid rhinorrhea, which can be repaired endoscopically during the same setting [7]. In this report, we describe a case of a large OF that was resected endoscopically without complications and without recurrence at the 2-year follow-up. 4. Conclusion Endoscopic resection of sinonasal ossifying fibromas is an excellent therapeutic option when done by a surgeon experienced in endoscopic sinonasal surgery. The tumorTs well-demarcated borders allow for complete resection and promote assurance of tumor-free margins, although recurrence is always possible. The additional advantage that the endoscopic provides in these cases is excellent postoperative surveillance for tumor recurrence. References [1] Marvel JB, Marsh MA, Catlin FI. Ossifying fibroma of the mid-face and paranasal sinuses: diagnostic and therapeutic considerations. Otolaryngol Head Neck Surg 1991;104:803 - 8. [2] Johnson LC, Yousefi M, Vinh TN, et al. Juvenile active ossifying fibroma: its nature, dynamics and origin. Acta Otolaryngol Suppl (Stockh) 1991;488:1 - 40. [3] London SD, Schlosser RJ, Gross CW. Endoscopic management of benign sinonasal tumors: a decade of experience. Am J Rhinol 2002; 16:221 - 7. [4] Vaidya AM, Chow JM, Goldberg K, et al. Juvenile aggressive ossifying fibroma presenting as an ethmoid sinus mucocele. Otolaryngol Head Neck Surg 1998;119:665 - 8. [5] Chong VFH, Tan LHC. Maxillary sinus ossifying fibroma. Am J Otolaryngol 1997;18:419 - 24. [6] Choi YC, Jeon EJ, Park YS. Ossifying fibroma arising in the right ethmoid sinus and nasal cavity. Int J Pediatr Otorhinolaryngol 2000; 54:159 - 62. [7] Brodish BN, Morgan CE, Sillers MJ. Endoscopic resection of fibroosseous lesions of the paranasal sinuses. Am J Rhinol 1999;13:111 - 6.