Original Investigation
Orbital Osteoma: Clinical Features and Management Options Leslie A. Wei, M.D.*, Nicholas A. Ramey, M.D.†, Vikram D. Durairaj, M.D., F.A.C.S. *‡, Vijay R. Ramakrishnan, M.D.‡, Augusto V. Cruz, M.D., Ph.D.§║¶, Peter J. Dolman, M.D., F.R.C.S.C.#, and Mark J. Lucarelli, M.D., F.A.C.S.† *Department of Ophthalmology, Oculoplastic and Orbital Surgery Service, University of Colorado Denver, Denver, Colorado; †Department of Ophthalmology, Oculoplastic, Facial Cosmetic, and Orbital Surgery Service, University of Wisconsin– Madison, Madison, Wisconsin; ‡Department of Otolaryngology and Head and Neck Surgery, University of Colorado Denver, Denver, Colorado, U.S.A.; §Oculoplastics and Orbit Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia; ║Oculoplastics Division, Wilmer Eye Institute, Johns Hopkins University, Baltimore, Maryland, U.S.A.; On leave of absence from the ¶Craniofacial Unit of the Department of Ophthalmology, Otorhinolaryngology and Head and Neck Surgery, School of Medicine of Ribeirão Preto–University of São Paulo, São Paulo, Brazil; and #Department of Ophthalmology, Oculoplastics, Lacrimal, and Orbital Surgery Service, University of British Columbia, Vancouver, British Columbia, Canada
Purpose: This study reviews the clinical presentation and management of 11 cases of sino-orbital osteoma. Methods: The medical records of patients with primary (originating from orbital bone) and secondary (originating from the paranasal sinuses) orbital osteoma from the academic practices of 4 surgeons (A.V.C., M.J.L., P.J.D., V.D.D.) were reviewed for clinical presentation and course, pathologic study, and radiologic reports . A Medline search of English-language literature on orbital osteomas was conducted for comparison with these findings. Results: Eleven cases of primary (1) and secondary (10) orbital osteoma were reviewed, with a mean follow up of 16 months. Seven patients were women. Ages ranged from 15–68 years, with a median of 40 years. Presenting complaints included slowly progressive globe displacement, palpable bony nodule, pain, and diplopia. Surgery was performed in 10 cases. Surgical approach varied according to location and size of each lesion and was performed in combination with otolaryngology and neurosurgery services as needed. Reconstruction included sculpting osteomatous bone to natural orbital contours, repair of orbital wall defects with implants, and obliteration of frontal sinus. Lesions demonstrated mixed compact, cancellous, and fibrous histologic subtypes. Conclusions: Osteomas are the most common tumor of the paranasal sinuses (noted in up to 3% of coronal CT images), but secondary extension in or primary involvement of the orbit is rare. A variety of surgical approaches led to successful outcomes in this series. Complete surgical removal is not always necessary, and partial sculpting may relieve symptoms and cause less surgical morbidity in selected cases. Accepted for publication September 15, 2013. Presented at the American Society of Ophthalmic Plastic and Reconstructive Surgery 2012 Fall Symposium, Chicago, Illinois, U.S.A., November 9, 2012. Supported in part by an unrestricted grant from Research to Prevent Blindness Inc., New York, New York, U.S.A., to Department of Ophthalmology and Visual Sciences, University of Wisconsin–Madison. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Mark J. Lucarelli, M.D., F.A.C.S., Department of Ophthalmology and Visual Sciences, University of Wisconsin–Madison, 2880 University Ave., Madison, WI 53705. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000039
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P
rimary orbital bone tumors comprise 0.6% to 2.0% of all orbital tumors.1 Of these, osteoma and fibrous dysplasia are most common.1 Osteomas are benign, slow-growing bone tumors that can occur in any bone, usually in the appendicular skeleton. They are the most common tumor of the paranasal sinuses, originating in decreasing frequency from the frontal, ethmoid, maxillary, and sphenoid sinuses.2 Osteomas are most commonly small and asymptomatic, being noted incidentally on up to 3% of head CT scans obtained for other indications.3 Secondary orbital extension from the paranasal sinuses has historically been considered uncommon, with an incidence ranging from 0.9% to 5.1%.4 However, in 1 case series, orbital extension was found in 24% of cases of ethmoid sinus osteomas.5 Primary orbital bone osteomas are very rare, usually appearing in the literature as case reports.6–10 Although unusual, orbital involvement can cause eye pain, diplopia, metamorphopsia, globe displacement, epiphora, dacryocystitis, orbital cellulitis, and vision loss.4,10,11 Nonorbital symptoms include sinusitis, headache, facial pain, and seizures.9,12 Due to relatively higher frequency in the paranasal sinuses, the natural history and postsurgical behavior of osteomas are well documented in the Otolaryngology literature.13–17 The primary objectives of this study were to highlight the clinical oculofacial manifestations of orbital osteomas and to explore the varied surgical strategies used to manage these lesions.
METHODS Medical records from the academic practices of 4 senior authors were reviewed retrospectively for primary (originating from orbital bone) and secondary (originating from the paranasal sinuses) orbital osteomas identified between January 1, 2000 and December 1, 2012. Clinical parameters reviewed included history and examination findings, imaging characteristics, management decision making, operative goals, postoperative course, and histopathology. Institutional Review of this retrospective case series was waived. The MEDLINE database was searched using PubMed for English-language articles using the search terms “orbital osteoma” and “paranasal sinus osteoma,” with emphasis on articles published within the last 5 years. Abstracts were reviewed for articles providing updates on osteoma pathophysiology, clinical behavior, and surgical approaches.
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RESULTS Eleven cases of orbital osteoma from the 4 academic practices were identified. One case was considered a primary orbital lesion, and 10 were secondary from the adjacent paranasal sinuses. Seven patients were women. Median age was 40 years (range 15–68). Mean follow up was 18 months (range 2 months–8 years). Forty-five percent of the patients presented with slowly progressive globe displacement. Other presenting complaints included a palpable bony nodule, eye pain, diplopia, blurry vision, and orbital emphysema (Table 1). Tumors were categorized by their location in the orbit and their presumed site of origin, that is, orbital bone, frontal sinus, ethmoid sinus, and frontoethmoidal region. Ten of 11 patients underwent surgery; the remaining case is still under close observation. The surgical approach to each case was customized to the size and location of the osteoma and to the specific goals of
the surgery (i.e., sculpting vs. complete excision). Of the 10 surgical cases, 5 patients underwent a coronal approach, 3 transcaruncular/transconjunctival, 1 combined upper eyelid crease and endoscopic, and 1 endonasal. Total resection was planned and achieved in 6 patients. Partial or near-total resection was achieved in 4 patients. In 1 patient, a second surgery was elected to remove residual osteoma from the paranasal sinuses. Surgical duration of time ranged from roughly 1 to 3 hours. Complications included recurrent frontal sinusitis, iatrogenic superior oblique paresis that eventually required surgical correction, deep superior sulcus, postoperative frontal sinusitis with late-stage resorption, and eventual loss of the anterior table of the frontal sinus. There were no cases of postoperative hemorrhage or wound dehiscence. Reconstruction techniques included repairing orbital wall defects with implants such as porous polyethylene, titanium mesh, and hydroxyapatite (5), sculpting osteomatous bone to natural orbital contours (4),
TABLE 1. Demographics and relevant clinical features of osteoma cases Patient Age Gender
Presenting complaint
Male Left superior globe displacement, palpable bony nodules along inferior rim
Proptosis, (mm)
1
53
2
18
Female Right medial orbital palpable nodule, eye pain
3
15
4
64
Female Left blurry vision and eye pain Female Left proptosis and inferior displacement, vertical diplopia
5
40
6
40
7
35
Female Left superior rim palpable nodule
—
Primary orbital
8
68
Female Right superior rim palpable nodule
1
Frontal
9
17
Male Right proptosis and inferior globe displacement
12
Frontal
10
19
0
11
44
Male Right upper eyelid emphysema during exercise Male OD pain
Female Right proptosis, diplopia on upgaze, and headaches Female Left proptosis
1
Origin
Surgical technique
Histopathology
Outcome and complications
Composite of woven Restoration of normal and lamellar eye position, no bone deposited in diplopia. Retained cortical pattern with maxillary sinus Haversianpolyp. like systems. Scant fibrous stroma. 1 mm Ethmoid 1) Transcaruncular approach, 2) Anastomosing Improved eye position. Modified Lynch incision. Total trabeculae of woven Self-limited resection bone, spaces filled postoperative with vascular loose diplopia in upgaze. connective tissue 2 Ethmoid Nasal endoscopy. Near -total Osteoma with Improved visual acuity (bilateral) resection. osteoblastic features and eye pain. 4 Frontal Combined eyelid crease incision — Restoration of normal and nasal endoscopy. Partial eye position and resection and frontal sinus resolution of fenestration. diplopia. 8 Frontal, Surgery deferred N/a N/a ethmoid 4 mm
2
Maxillary Transconjunctival swinging eyelid approach. Near-total resection
Frontal
Coronal approach. Titanium mesh reconstruction. Total resection. Coronal approach. Titanium mesh reconstruction. Total resection. Coronal approach, reconstruction with medpor and hydroxyapatite. Total resection
Coronal approach, obliteration of frontal sinus with temporalis/ galea flap, reconstruction with titanium mesh. Total resection. Ethmoid Combined transcaruncular and nasal endoscopic approach. Total resection. Frontal, Coronal approach. ethmoid Reconstruction with titanium (bilateral) mesh. Outer table of frontal sinus replaced. Near-total resection.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
— — —
—
Restoration of normal eye position. Deep superior sulcus. Improved orbital rim contour. Improved orbital rim contour. Postoperative torticollis from superior oblique paresis requiring plication, then symptom-free. Restoration of normal eye position.
—
Resolution of symptoms.
—
Recurrent frontal sinusitis. Eventual loss of outer table of both frontal sinuses. Osteoma recurrence in right ethmoid.
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obliterating the frontal sinus, and inserting temporalis/galeal flaps (1). Histopathology was available for 3 cases and demonstrated mixed compact, cancellous, and fibrous subtypes (Table 1). A conceptual literature review was performed using PubMed, focusing particularly on articles published within the last 5 years. Pertinent abstracts were identified and reviewed based on the search criteria stated in the methods. Forty-four full text manuscripts were reviewed, addressing the incidence, natural history, presenting symptoms, surgical management, postoperative course, and associated syndromes for osteomas of the facial skeleton. Twenty-six of these presented cases of osteomas specifically involving the orbit (Table 2). Most orbital osteomas occurred in patients in their fourth and fifth decades. Surgical approaches described include endoscopic, coronal, transcaruncular, transblepharoplasty incision, and combined. Over the last 5 years, most surgical cases reported in the literature were completely excised endoscopically. Those that were incompletely resected typically underwent a second procedure to remove residual tumor. Recurrence was extremely rare, with only 1 study reporting recurrent frontal sinus osteoma 7 years after initial surgery. Selected Cases. Case 1. This case highlights subtotal resection as a safe and effective management option for orbital osteomas. A 53-year-old man
presented with a 6-month history of left hyperglobus and mass effect on the inferior orbital rim. Best corrected visual acuity (BCVA) was 20/25, and the remainder of the examination was normal with the exception of 1 mm left exophthalmos and a palpable bony mass of the left inferior orbital rim (Fig. 1). Imaging showed a 2.2 × 2.1 × 1.6 cm osteoma of the left orbital floor abutting the left globe inferiorly and displacing the orbital soft tissues superiorly. The patient underwent planned near-total resection of the lesion by sculpting the osteoma flush with normal contour of the floor and inferior orbital rim via a lower eyelid transconjunctival approach with lateral canthotomy and cantholysis. Histopathology showed a composite of woven and lamellar bone, deposited in a cortical pattern with Haversian-like systems with scant fibrous stroma. The portion of the osteoma involving the zygoma outside the orbit was left intact. On follow-up month 3, the patient’s eye was in normal position, motility was intact, and he was orthophoric, with BCVA 20/15. Case 2. This case demonstrates the potentially protean nature of sinoorbital osteomas and the possible systemic association with Gardner syndrome. A 15-year-old woman presented with progressively worsening blurry vision and metamorphopsia of the left eye for 1 year. She had also developed headaches and a pressure sensation behind her left eye over several months prior to presentation. On examination, BCVA OS
TABLE 2. Summary of selected osteoma case reports and series with emphasis on the years 2007–2012 Study
Year
n
2012
1
15
F
Frontal sinus
External, multistage
None
Kayaci et al.8 Koktekir et al.11 Nicolotti et al.31
2012 2012 2012
1 1 1
80 16 62
F M M
Sphenoid Ethmoid Frontal sinus
None None None
Pons et al.5
2012
25
44
Turri-Zanoni et al.28 2012
60
44.1
Yazici et al.23
2012
3
18
Coronal Endoscopic Direct frontal excision w/titanium 4 coronal, 19 endoscopic, 2 combined 4 external, 31 endo, 25 combined External
Friedrich9
2009
1
53
Livaoĝlu et al.29
2009
1
55
Miman et al.38
2009
1
28
Seiberling et al.16
2009
23
47
Kashkouli et al.7 Gerbrandy et al.39 Andaloussi et al.40
2008 2007 2006
1 1 3
22 39 26.3
Selva et al.1 Sibony et al.41 Eppley et al.42 Ma’luf et al.43 Naraghi et al.44 Selva et al.15 Becelli et al.6 Sires et al.10 Alper et al.45 Kobayashi et al.46 Naidu et al.47
2004 2004 2003 2003 2003 2003 2002 1999 1998 1996 1987
11 1 2 1 1 1 2 1 1 2 1
Ns 54 39.5 27 42 28 39.5 15 22 2 15
Albert et al.48
1982
1
Fan et al.
12
Avg age
74
Gender
Osteoma origin
Surgical technique
16 M, 9 F Ethmoid sinus (24% Orbit) 33 M, 27 F Frontal/ethmoid (10% Orbit) 3M Ethmoid/ frontoethmoid F Primary Orbit (Floor) Infraorbital with Ethisorb
Complications
None
Outcome Continued reconstruction of lower face Sx improved Sx resolved Sx resolved, forehead numbness No recurrence
Persistent Complete resection in diplopia (1) all but 2 cases None Sx resolved, no recurrence None Resolved sx and no recurrence M Primary orbit (roof) Blepharoplasty incision None Resolved sx and no recurrence F Ethmoid sinus Endoscopic None Sx resolved, mild enophthalmos 11 M, 12 F Frontal sinus (4.3% 13 endo, 10 external, Frontal sinus Sx resolved, no Orbit) 2 combined narrowing (1) recurrence M Primary orbit (roof) Coronal None Sx resolved F Ethmoid Combined None Sx resolved 3F Frontal/ethmoid External Ptosis and Sx resolved diplopia (1) Ns Frontal/ethmoid Ns (only 9 required surgery) Ns 1 recurrence F Ethmoid External None Sx resolved 1 M, 1 F Frontal sinus External None Sx resolved M Ethmoid Combined None Sx resolved M Ethmoid Endoscopic None Sx resolved F Frontal/ethmoid Endoscopic and eyelid crease None Sx resolved 1 M, 1 F Primary orbit Coronal, subciliary None Complete excision M Ns Blepharoplasty incision None Sx resolved M Ethmoid Coronal None Sz resolved 1 M, 1 F Frontal/maxillary Coronal None Sx resolved M Primary orbit (medial Coronal None Sx resolved wall) F Ethmoid/maxillary External None Sx resolved
Ns, not specified; endo, endoscopic; sx, symptoms. A series was included only if it contained orbital cases.
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FIG. 1. Top Left: Case 1, 53-year-old man with left hyperglobus and palpable inferior rim deformity due to a sino-orbital osteoma. Top Right: Axial CT showing left maxillary sinus/orbital floor osteoma. Bottom Left: Intraoperative photographs after partial resection and sculpting of the floor and inferior rim. Bottom right: Hematoxylin-eosin stain of specimen. Note composition of mostly mature-appearing lamellar bone with Haversian-like systems. was 20/80 with no motility deficits or afferent pupillary defect. She had 2 mm of left telecanthus and 2 mm of proptosis (Fig. 2). Funduscopic examination revealed choroidal folds in the macula and indentation of the medial aspect of the globe. A large left ethmoidal osteoma impinging on the medial rectus and small right ethmoidal osteoma were found on imaging. The patient underwent image-guided transnasal endoscopic resection of both osteomas. At follow-up month 4, the patient’s BCVA OS was 20/40, and she reported no further headaches or eye pain. Her left fovea returned to its normal contour with resolution of the choroidal folds, although she continued to have a mild epiretinal membrane. A small amount of residual tumor was left at the attachment site along the left frontal recess and cribriform plate. The patient was also referred for genetic evaluation for Gardner syndrome, as her father had a history of multiple colon polyps. At 1-year follow up, the patient was asymptomatic and did not show any growth of the residual osteoma on nasal endoscopy or CT scan. Neither she nor her family members have undergone further work-up for Gardner syndrome. Case 3. This case demonstrates the importance of a multidisciplinary surgical approach to large sino-orbital osteomas. A 17-year-old man presented with severe right inferior globe displacement and 12 mm of right proptosis (Fig. 3). On imaging, he was found to have a large right frontal sinus osteoma with secondary extension in the orbit. The patient underwent complete resection via coronal approach with an osteoplastic flap. The anterior table of the frontal sinuses was noted to be thin during the initial resection, and the frontal sinus was obliterated using a temporalis/galea flap to increase the chance of survival of the anterior table. The orbital roof was reconstructed with porous polyethylene titanium mesh (Stryker, Kalamazoo, MI, U.S.A.). No complications were noted at 4-month follow up, and the patient had resolution of proptosis and no new visual complaints. Case 4. This case also demonstrates the viability of partial resection as a treatment option in selected patients. An 18-year-old woman presented with eye pain and a right medial orbital palpable nodule for 1 year. On examination, BCVA was 20/25 in the affected eye, and she had a superomedial nonmobile mass with 1 mm proptosis. She was found to have a 2.6 × 2.2 × 1.8 cm right ethmoid osteoma extending in the right orbit, bulging up to the lateral lamella of the cribiform plate and causing frontal recess obstruction. She underwent partial resection/drilling
of the orbital component of the osteoma via transcaruncular approach to relieve symptoms from mass effect. The ethmoidal component of the osteoma was initially left undisturbed, given its proximity to the cribriform plate. However, she continued to have frontal recess obstruction and underwent a second surgery for complete excision via a modified Lynch incision. At the time of excision, she also underwent medial wall reconstruction with a porous polyethylene implant. Pathologic study of the resected bone fragments showed anastomosing trabeculae of woven bone and spaces filled with vascular loose connective tissue. At 2-month follow up, her proptosis was completely resolved, and symptoms were improved. She had mild diplopia in upgaze only.
DISCUSSION Sino-orbital osteomas are found incidentally on 1% of plain sinus radiographs and 3% of CT scans and are often small and asymptomatic.3,4,18 Given their slow growth rate (average 1.6 mm/year), asymptomatic or incidentally discovered lesions are typically observed.19 Approximately 5% of osteomas become symptomatic or require surgery.4 Osteomas may be more common in men, with the reported male to female ratio ranging from 1:1 to 2.4:1,2 although this predilection was not seen in this series. The differential diagnosis for orbital osteoma includes benign entities such as ossifying fibroma, fibrous dysplasia, osteoblastoma, and malignant neoplasms such as osteosarcoma and osteoblastic metastases.1 In the head and neck, osteomas are most commonly found in the paranasal sinuses—classically the frontal sinus—although some case series have found ethmoid osteomas to be more prevalent.2,20 Ethmoid sinus osteomas often originate from the lateral portion of ethmoid sinus roof and extend toward the orbit and also toward the frontal sinus.5 Grossly, sino-orbital osteomas have a whitish coloration and a smooth or bosselated surface. Histologically, osteomas exhibit variable patterns, most commonly mature lamellar bone with trabeculae.4 Several classification schemes based on histology and morphology have been used to describe osteomas. One common scheme differentiates lesions in ivory (compact), mature (cancellous), and mixed ivory/mature subtypes based on histology.21
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FIG. 2. A, Case 2, 18-year-old woman with left telecanthus, proptosis, and lateral globe displacement from an ethmoid osteoma. B, Fundus photograph showing macular choroidal folds. C, Coronal CT showing large left ethmoidal osteoma displacing the globe laterally and causing proptosis. Note the thick calcified rim and polypoid configuration. D, Hematoxylin-eosin stain of cortical area of specimen. E, Hematoxylin-eosin stain of trabecular area of specimen. F, Medium magnification of central osteoblastoma-like area of specimen.
Others suggest this classification scheme has little clinical relevance and instead describe lesions morphologically based on grossly organized concentric zones of bone maturation.1 The “osteoma with osteoblastoma-like features” histologic subtype is marked by interanastomosing trabeculae and fibrovascular stroma surrounded by osteoblasts and osteoclasts.22,23 Osteomas with osteoblastoma-like features are more likely to exhibit extracavitary extension, cause visual symptoms, and be incompletely excised; however, they may be less likely to recur than other osteomas.22 They also have a distinct radiographic zonal appearance, with lower density osteoblastoma-like material at the core and denser mature bone in the periphery.23 On imaging, osteomas appear often as dense, sclerotic, well-defined masses forming a polypoid configuration. CT is the imaging modality of choice, given its excellent rendering of bony structures.23 MRI may give a falsely aggressive appearance to osteoid osteoma. More accurate radiologic
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diagnoses are made by CT as opposed to MRI.24 However, in the case of possible intracranial extension, MRI is also recommended.25 Indications for surgical intervention include vision loss or optic neuropathy, eye pain, proptosis or globe displacement, diplopia, palpable defect, postobstructive sinus disease, chronic headaches, or cosmetic deformity.4,26 Georgalas et al.27 proposed treatment when the following criteria are met: 1) Osteomas associated with symptoms after all other explanations for the symptoms have been excluded, 2) Large (extending to >50% of the frontal sinus) or growing osteomas, as seen on serial CTs, and 3) Osteomas associated with current complications (mucocele, orbital symptoms, neurologic symptoms, external deformity) or imminent (complete obstruction of the frontal recess or intraorbital/intracranial extension) complications. Because growth appears to stem outward from a central core, partial resection that includes removal of this core is also regarded to be a viable alternative to complete resection.28 Several surgical approaches to primary and secondary orbital osteomas have been described.28–30 A transconjunctival inferior fornix approach is useful for inferior orbital lesions. Anterior orbitotomy by lateral canthotomy with swinging lower eyelid approach provides exposure for inferior anterior orbital lesions. Superior lesions may be accessed via an upper eyelid crease incision, but extensive lesions may also require the wider exposure afforded by a coronal incision. The coronal approach is useful for giant frontal osteomas (see Case 3), requiring reconstruction for optimal functional and cosmetic results.31 A transcaruncular or medial canthal cutaneous approach, with or without nasal endoscopy, may be suitable for superomedial osteomas. The endoscopic approach is preferred for many ethmoid osteomas and can be used judiciously for frontal sinus osteomas and even selected osteomas with intraorbital extension.5,25,28 Aspects to consider for endoscopic removal of frontal sinus osteomas include 1) location of base of attachment, 2) location in relation to a virtual sagittal plane through the lamina papyracea, and 3) relative size of lesion in comparison with the frontal recess.26 Many frontal sinus osteomas can be removed endoscopically with the exception of those with a superior pedicle, far lateral location, or large size compared with the frontal recess.16,26,32 For complete resection of osteomas, greater surgical exposure is necessary than for resection of similarly sized soft-tissue tumors. Because reconstructions depend on intraoperative interpretation of the bony defect, and the natural and highly variable topography of the orbit, no single set of criteria (for defect size or location) is applicable. The choice of a particular approach depends not only on the size and location of the osteoma but also the experience of the surgeon. Autografting, alloplastic implantation, and sculpting of the osteomatous bone represent effective reconstruction techniques. Although developmental abnormality, trauma, and infection/chronic inflammation have been proposed as possible mechanisms, the etiology of osteomas remains unknown.1,6,14 Most osteomas are sporadic. However, they may be associated with Gardner syndrome (phenotypic variant of Familial Adenomatosis Polyposis or FAP), an inherited colonic adenomatosis in which the incidence of colon adenocarcinoma is 100%. Other manifestations of Gardner syndrome include extracolonic malignancies such as papillary thyroid carcinoma, mesenteric fibromatosis, hepatoblastoma, and gastric carcinoma.33,34 Gardner syndrome is also characterized by benign extracolonic lesions such as nasal angiofibromas, adrenal adenomas, and congenital hyperplasia of the retinal pigment
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FIG. 3. A, Case 3, 17-year-old man with severe right proptosis and inferior globe displacement from frontal sinus osteoma. B, Coronal CT showing large frontal sinus osteoma with orbital extension and globe displacement. C, Postoperative photograph showing improvement in eye position and reduced proptosis. D, Intraoperative photograph showing bicoronal flap and unroofed frontal sinus. E, Intraoperative photograph showing completely resected right frontal sinus osteoma. F, Intraoperative photograph after obliteration of the frontal sinus with temporalis/galea flap and porous polyethylene titanium mesh roof reconstruction.
epithelium (CHRPE), which are usually multiple and bilateral.34,35 On average, clinically evident osteomas are found 17 years before a diagnosis of Gardner syndrome is made.36 As both the colonic and extracolonic manifestations of this syndrome are life threatening, often at a young age, the oculoplastic surgeon can impact outcome by initiating a work-up for early detection. Patients with osteoma and suspicion of Gardner syndrome (positive family history, gastrointestinal symptoms, and/or extracolonic manifestations) should have a dilated funduscopic exam, genetics consult with possible testing for adenomatous polyposis coli mutations, and Gastroenterology consult for possible flexible sigmoidoscopy or colonoscopy.36,37 While this series contains several paranasal sinus osteomas with orbital extension, it is limited by relatively few cases of primary orbital osteoma—a function of this lesion’s extremely low prevalence. Follow-up time (mean 18 months) was also limited in many of these cases. It is possible that additional morbidity related to wide/extensive excision or recurrence may be discovered with greater follow up. Given the slow general growth and benign nature of these tumors, subtotal resection is reasonable when complete excision would create disproportionate morbidity or significantly complicate surgery or reconstruction. Patients should be monitored for recurrence or growth of the residual tumor with annual clinical exams and biannual imaging. If clinical and radiographic stability is reached after a period of 5 years, imaging can be performed on an as-needed basis for new symptoms or examination findings. Drawing from 4 separate academic institutions, this study was unable to control for variations in clinical recording, surgical technique, histologic review, and follow up among senior
authors. Also, the geographically and culturally diverse populations contributing these cases make discerning common etiologic risk factors difficult.
CONCLUSIONS Although osteomas are the most common bony tumor of the paranasal sinuses, orbital extension and primary orbital osteoma are rare and can present variably. Radiographic, gross, and histologic variations appear to have little impact on clinical behavior. A multidisciplinary surgical team may be required, potentially using both endoscopic and open approaches. Surgical goals may include partial or complete resection, in addition to reconstruction by bony sculpting and/or implantation of alloplastic implant materials. Partial resection is a viable and potentially preferable alternative when complete resection risks excessive morbidity or unnecessary surgical complexity. Overall, clinical outcomes of symptomatic lesions are favorable.
REFERENCES 1. Selva D, White VA, O’Connell JX, et al. Primary bone tumors of the orbit. Surv Ophthalmol 2004;49:328–42. 2. Namdar I, Edelstein DR, Huo J, et al. Management of osteomas of the paranasal sinuses. Am J Rhinol 1998;12:393–8. 3. Erdogan N, Demir U, Songu M, et al. A prospective study of paranasal sinus osteomas in 1,889 cases: changing patterns of localization. Laryngoscope 2009;119:2355–9. 4. Mansour AM, Salti H, Uwaydat S, et al. Ethmoid sinus osteoma presenting as epiphora and orbital cellulitis: case report and literature review. Surv Ophthalmol 1999;43:413–26. 5. Pons Y, Blancal JP, Vérillaud B, et al. Ethmoid sinus osteoma: diagnosis and management. Head Neck 2013;35:201–4.
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6. Becelli R, Santamaria S, Saltarel A, et al. Endo-orbital osteoma: two case reports. J Craniofac Surg 2002;13:493–6. 7. Kashkouli MB, Khalatbari MR, Yahyavi T, et al. Primary endoorbital osteoid osteoma. Orbit 2008;27:211–3. 8. Kayaci S, Kanat A, Gucer H, et al. Primary osteoma of the orbit with atypical facial pain: case report and literature review. Turk Neurosurg 2012;22:389–92. 9. Friedrich RE. Long-term follow-up control of pedunculated orbital floor osteoma becoming symptomatic by atypical facial pain. In Vivo 2009;23:117–21. 10. Sires BS, Benda PM, Stanley RB Jr, et al. Orbital osteoid osteoma. Arch Ophthalmol 1999;117:414–5. 11. Koktekir BE, Ozturk K, Gedik S, et al. Giant ethmoido-orbital osteoma presenting with dacryocystitis and metamorphopsia. J Craniofac Surg 2012;23:e390–2. 12. Fan KL, Ghadjar K, Yuan JT, et al. Giant cranial osteoma: successful staged excision of the largest reported. J Craniofac Surg 2012;23:e480–2. 13. Buyuklu F, Akdogan MV, Ozer C, et al. Growth characteristics and clinical manifestations of the paranasal sinus osteomas. Otolaryngol Head Neck Surg 2011;145:319–23. 14. Eller R, Sillers M. Common fibro-osseous lesions of the paranasal sinuses. Otolaryngol Clin North Am 2006;39:585–600, x. 15. Selva D, Chen C, Wormald PJ. Frontoethmoidal osteoma: a stereotactic-assisted sino-orbital approach. Ophthal Plast Reconstr Surg 2003;19:237–8. 16. Seiberling K, Floreani S, Robinson S, et al. Endoscopic management of frontal sinus osteomas revisited. Am J Rhinol Allergy 2009;23:331–6. 17. Pagella F, Pusateri A, Matti E, et al. Transnasal endoscopic approach to symptomatic sinonasal osteomas. Am J Rhinol Allergy 2012;26:335–9. 18. Vowles RH, Bleach NR. Frontoethmoid osteoma. Ann Otol Rhinol Laryngol 1999;108:522–4. 19. Koivunen P, Löppönen H, Fors AP, et al. The growth rate of osteomas of the paranasal sinuses. Clin Otolaryngol Allied Sci 1997;22:111–4. 20. Çelenk F, Baysal E, Karata ZA, et al. Paranasal sinus osteomas. J Craniofac Surg 2012;23:e433–7. 21. Fu YS, Perzin KH. Non-epithelial tumors of the nasal cavity, paranasal sinuses, and nasopharynx. A clinicopathologic study. II. Osseous and fibro-osseous lesions, including osteoma, fibrous dysplasia, ossifying fibroma, osteoblastoma, giant cell tumor, and osteosarcoma. Cancer 1974;33:1289–305. 22. McHugh JB, Mukherji SK, Lucas DR. Sino-orbital osteoma: a clinicopathologic study of 45 surgically treated cases with emphasis on tumors with osteoblastoma-like features. Arch Pathol Lab Med 2009;133:1587–93. 23. Yazici Z, Yazici B, Yalcinkaya U, et al. Sino-orbital osteoma with osteoblastoma-like features: case reports. Neuroradiology 2012;54:765–9. 24. Hosalkar HS, Garg S, Moroz L, et al. The diagnostic accuracy of MRI versus CT imaging for osteoid osteoma in children. Clin Orthopaedics Rel Res 2005:171–7. 25. Rokade A, Sama A. Update on management of frontal sinus osteomas. Curr Opin Otolaryngol Head Neck Surg 2012;20:40–4. 26. Chiu AG, Schipor I, Cohen NA, et al. Surgical decisions in the management of frontal sinus osteomas. Am J Rhinol 2005;19:191–7. 27. Georgalas C, Goudakos J, Fokkens WJ. Osteoma of the skull base and sinuses. Otolaryngol Clin North Am 2011;44:875–90, vii.
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28. Turri-Zanoni M, Dallan I, Terranova P, et al. Frontoethmoidal and intraorbital osteomas: exploring the limits of the endoscopic approach. Arch Otolaryngol Head Neck Surg 2012;138: 498–504. 29. Livaoĝlu M, Cakir E, Karaçal N. Large orbital osteoma arising from orbital roof: Excision through an upper blepharoplasty incision. Orbit 2009;28:200–2. 30. Kim YJ, Kim DY, Jun YJ, et al. Ethmoid sinus osteoma associated with blow-out fracture. J Craniofac Surg 2012;23:477–9. 31. Nicolotti M, Grivetto F, Brucoli M, et al. Direct access to a frontal sinus osteoma and reconstruction of the orbital roof displaced by the lesion by titanium mesh. J Craniofac Surg 2012;23: e364–6. 32. Timperley DG, Banks C, Robinson D, et al. Lateral frontal sinus access in endoscopic skull-base surgery. Int Forum Allergy Rhinol 2011;1:290–5. 33. Alexander AA, Patel AA, Odland R. Paranasal sinus os teomas and Gardner’s syndrome. Ann Otol Rhinol Laryngol 2007;116:658–62. 34. Harned RK, Buck JL, Olmsted WW, et al. Extracolonic manifestations of the familial adenomatous polyposis syndromes. AJR Am J Roentgenol 1991;156:481–5. 35. Touriño R, Conde-Freire R, Cabezas-Agrícola JM, et al. Value of the congenital hypertrophy of the retinal pigment epithelium in the diagnosis of familial adenomatous polyposis. Int Ophthalmol 2004;25:101–12. 36. Bülow S. Incidence of associated diseases in familial polyposis coli. Semin Surg Oncol 1987;3:84–7. 37. Kerr SE, Thomas CB, Thibodeau SN, et al. APC germline mutations in individuals being evaluated for familial adenomatous polyposis: a review of the Mayo Clinic experience with 1591 consecutive tests. J Mol Diagn 2013;15:31–43. 38. Miman MC, Bayindir T, Akarcay M, et al. Endoscopic removal technique of a huge ethmoido-orbital osteoma. J Craniofac Surg 2009;20:1403–6. 39. Gerbrandy SJ, Saeed P, Fokkens WJ. Endoscopic and trans fornix removal of a giant orbital-ethmoidal osteoma. Orbit 2007;26:299–301. 40. Andaloussi BI, Touiza E, Bhallil S, et al. Orbital osteoma: three case reports. Bull Soc Belge Ophtalmol. 2006;300:73–9. 41. Sibony P, Shindo M. Orbital osteoma with gaze-evoked amaurosis. Arch Ophthalmol 2004;122:788. 42. Eppley BL, Kim W, Sadove AM. Large osteomas of the cranial vault. J Craniofac Surg 2003;14:97–100. 43. Ma’luf RN, Ghazi NG, Zein WM, et al. Orbital osteoma arising adjacent to a foreign body. Ophthal Plast Reconstr Surg 2003;19:327–30. 44. Naraghi M, Kashfi A. Endonasal endoscopic resection of ethmoido-orbital osteoma compressing the optic nerve. Am J Otolaryngol 2003;24:408–12. 45. Alper M, Gürler T, Totan S, et al. Intraorbital osteoma and surgical strategy. J Craniofac Surg 1998;9:464–7. 46. Kobayashi S, Ohmori K, Akizuki T. Combined use of an orbital osteotomy and living bone graft in the treatment of huge ossifying tumors involving the orbit. J Craniofac Surg 1996;7:156–9. 47. Naidu MR, Reddy DR, Reddy PK, et al. Intra orbital osteoma–a case report. Indian J Ophthalmol 1987;35:214–5. 48. Albert DM, Ni C, Sebag J, et al. Rare orbital tumors. Int Ophthalmol Clin 1982;22:183–205.
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Orbital Osteoma: Clinical Features and Management Options Leslie A. Wei, M.D.*, Nicholas A. Ramey, M.D.†, Vikram D. Durairaj, M.D., F.A.C.S. *‡, Vijay R. Ramakrishnan, M.D.‡, Augusto V. Cruz, M.D., Ph.D.§║¶, Peter J. Dolman, M.D., F.R.C.S.C.#, and Mark J. Lucarelli, M.D., F.A.C.S.† *Department of Ophthalmology, Oculoplastic and Orbital Surgery Service, University of Colorado Denver, Denver, Colorado; †Department of Ophthalmology, Oculoplastic, Facial Cosmetic, and Orbital Surgery Service, University of Wisconsin– Madison, Madison, Wisconsin; ‡Department of Otolaryngology and Head and Neck Surgery, University of Colorado Denver, Denver, Colorado, U.S.A.; §Oculoplastics and Orbit Division, King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia; ║Oculoplastics Division, Wilmer Eye Institute, Johns Hopkins University, Baltimore, Maryland, U.S.A.; On leave of absence from the ¶Craniofacial Unit of the Department of Ophthalmology, Otorhinolaryngology and Head and Neck Surgery, School of Medicine of Ribeirão Preto–University of São Paulo, São Paulo, Brazil; and #Department of Ophthalmology, Oculoplastics, Lacrimal, and Orbital Surgery Service, University of British Columbia, Vancouver, British Columbia, Canada
Purpose: This study reviews the clinical presentation and management of 11 cases of sino-orbital osteoma. Methods: The medical records of patients with primary (originating from orbital bone) and secondary (originating from the paranasal sinuses) orbital osteoma from the academic practices of 4 surgeons (A.V.C., M.J.L., P.J.D., V.D.D.) were reviewed for clinical presentation and course, pathologic study, and radiologic reports . A Medline search of English-language literature on orbital osteomas was conducted for comparison with these findings. Results: Eleven cases of primary (1) and secondary (10) orbital osteoma were reviewed, with a mean follow up of 16 months. Seven patients were women. Ages ranged from 15–68 years, with a median of 40 years. Presenting complaints included slowly progressive globe displacement, palpable bony nodule, pain, and diplopia. Surgery was performed in 10 cases. Surgical approach varied according to location and size of each lesion and was performed in combination with otolaryngology and neurosurgery services as needed. Reconstruction included sculpting osteomatous bone to natural orbital contours, repair of orbital wall defects with implants, and obliteration of frontal sinus. Lesions demonstrated mixed compact, cancellous, and fibrous histologic subtypes. Conclusions: Osteomas are the most common tumor of the paranasal sinuses (noted in up to 3% of coronal CT images), but secondary extension in or primary involvement of the orbit is rare. A variety of surgical approaches led to successful outcomes in this series. Complete surgical removal is not always necessary, and partial sculpting may relieve symptoms and cause less surgical morbidity in selected cases. Accepted for publication September 15, 2013. Presented at the American Society of Ophthalmic Plastic and Reconstructive Surgery 2012 Fall Symposium, Chicago, Illinois, U.S.A., November 9, 2012. Supported in part by an unrestricted grant from Research to Prevent Blindness Inc., New York, New York, U.S.A., to Department of Ophthalmology and Visual Sciences, University of Wisconsin–Madison. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Mark J. Lucarelli, M.D., F.A.C.S., Department of Ophthalmology and Visual Sciences, University of Wisconsin–Madison, 2880 University Ave., Madison, WI 53705. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000039
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P
rimary orbital bone tumors comprise 0.6% to 2.0% of all orbital tumors.1 Of these, osteoma and fibrous dysplasia are most common.1 Osteomas are benign, slow-growing bone tumors that can occur in any bone, usually in the appendicular skeleton. They are the most common tumor of the paranasal sinuses, originating in decreasing frequency from the frontal, ethmoid, maxillary, and sphenoid sinuses.2 Osteomas are most commonly small and asymptomatic, being noted incidentally on up to 3% of head CT scans obtained for other indications.3 Secondary orbital extension from the paranasal sinuses has historically been considered uncommon, with an incidence ranging from 0.9% to 5.1%.4 However, in 1 case series, orbital extension was found in 24% of cases of ethmoid sinus osteomas.5 Primary orbital bone osteomas are very rare, usually appearing in the literature as case reports.6–10 Although unusual, orbital involvement can cause eye pain, diplopia, metamorphopsia, globe displacement, epiphora, dacryocystitis, orbital cellulitis, and vision loss.4,10,11 Nonorbital symptoms include sinusitis, headache, facial pain, and seizures.9,12 Due to relatively higher frequency in the paranasal sinuses, the natural history and postsurgical behavior of osteomas are well documented in the Otolaryngology literature.13–17 The primary objectives of this study were to highlight the clinical oculofacial manifestations of orbital osteomas and to explore the varied surgical strategies used to manage these lesions.
METHODS Medical records from the academic practices of 4 senior authors were reviewed retrospectively for primary (originating from orbital bone) and secondary (originating from the paranasal sinuses) orbital osteomas identified between January 1, 2000 and December 1, 2012. Clinical parameters reviewed included history and examination findings, imaging characteristics, management decision making, operative goals, postoperative course, and histopathology. Institutional Review of this retrospective case series was waived. The MEDLINE database was searched using PubMed for English-language articles using the search terms “orbital osteoma” and “paranasal sinus osteoma,” with emphasis on articles published within the last 5 years. Abstracts were reviewed for articles providing updates on osteoma pathophysiology, clinical behavior, and surgical approaches.
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RESULTS Eleven cases of orbital osteoma from the 4 academic practices were identified. One case was considered a primary orbital lesion, and 10 were secondary from the adjacent paranasal sinuses. Seven patients were women. Median age was 40 years (range 15–68). Mean follow up was 18 months (range 2 months–8 years). Forty-five percent of the patients presented with slowly progressive globe displacement. Other presenting complaints included a palpable bony nodule, eye pain, diplopia, blurry vision, and orbital emphysema (Table 1). Tumors were categorized by their location in the orbit and their presumed site of origin, that is, orbital bone, frontal sinus, ethmoid sinus, and frontoethmoidal region. Ten of 11 patients underwent surgery; the remaining case is still under close observation. The surgical approach to each case was customized to the size and location of the osteoma and to the specific goals of
the surgery (i.e., sculpting vs. complete excision). Of the 10 surgical cases, 5 patients underwent a coronal approach, 3 transcaruncular/transconjunctival, 1 combined upper eyelid crease and endoscopic, and 1 endonasal. Total resection was planned and achieved in 6 patients. Partial or near-total resection was achieved in 4 patients. In 1 patient, a second surgery was elected to remove residual osteoma from the paranasal sinuses. Surgical duration of time ranged from roughly 1 to 3 hours. Complications included recurrent frontal sinusitis, iatrogenic superior oblique paresis that eventually required surgical correction, deep superior sulcus, postoperative frontal sinusitis with late-stage resorption, and eventual loss of the anterior table of the frontal sinus. There were no cases of postoperative hemorrhage or wound dehiscence. Reconstruction techniques included repairing orbital wall defects with implants such as porous polyethylene, titanium mesh, and hydroxyapatite (5), sculpting osteomatous bone to natural orbital contours (4),
TABLE 1. Demographics and relevant clinical features of osteoma cases Patient Age Gender
Presenting complaint
Male Left superior globe displacement, palpable bony nodules along inferior rim
Proptosis, (mm)
1
53
2
18
Female Right medial orbital palpable nodule, eye pain
3
15
4
64
Female Left blurry vision and eye pain Female Left proptosis and inferior displacement, vertical diplopia
5
40
6
40
7
35
Female Left superior rim palpable nodule
—
Primary orbital
8
68
Female Right superior rim palpable nodule
1
Frontal
9
17
Male Right proptosis and inferior globe displacement
12
Frontal
10
19
0
11
44
Male Right upper eyelid emphysema during exercise Male OD pain
Female Right proptosis, diplopia on upgaze, and headaches Female Left proptosis
1
Origin
Surgical technique
Histopathology
Outcome and complications
Composite of woven Restoration of normal and lamellar eye position, no bone deposited in diplopia. Retained cortical pattern with maxillary sinus Haversianpolyp. like systems. Scant fibrous stroma. 1 mm Ethmoid 1) Transcaruncular approach, 2) Anastomosing Improved eye position. Modified Lynch incision. Total trabeculae of woven Self-limited resection bone, spaces filled postoperative with vascular loose diplopia in upgaze. connective tissue 2 Ethmoid Nasal endoscopy. Near -total Osteoma with Improved visual acuity (bilateral) resection. osteoblastic features and eye pain. 4 Frontal Combined eyelid crease incision — Restoration of normal and nasal endoscopy. Partial eye position and resection and frontal sinus resolution of fenestration. diplopia. 8 Frontal, Surgery deferred N/a N/a ethmoid 4 mm
2
Maxillary Transconjunctival swinging eyelid approach. Near-total resection
Frontal
Coronal approach. Titanium mesh reconstruction. Total resection. Coronal approach. Titanium mesh reconstruction. Total resection. Coronal approach, reconstruction with medpor and hydroxyapatite. Total resection
Coronal approach, obliteration of frontal sinus with temporalis/ galea flap, reconstruction with titanium mesh. Total resection. Ethmoid Combined transcaruncular and nasal endoscopic approach. Total resection. Frontal, Coronal approach. ethmoid Reconstruction with titanium (bilateral) mesh. Outer table of frontal sinus replaced. Near-total resection.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
— — —
—
Restoration of normal eye position. Deep superior sulcus. Improved orbital rim contour. Improved orbital rim contour. Postoperative torticollis from superior oblique paresis requiring plication, then symptom-free. Restoration of normal eye position.
—
Resolution of symptoms.
—
Recurrent frontal sinusitis. Eventual loss of outer table of both frontal sinuses. Osteoma recurrence in right ethmoid.
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obliterating the frontal sinus, and inserting temporalis/galeal flaps (1). Histopathology was available for 3 cases and demonstrated mixed compact, cancellous, and fibrous subtypes (Table 1). A conceptual literature review was performed using PubMed, focusing particularly on articles published within the last 5 years. Pertinent abstracts were identified and reviewed based on the search criteria stated in the methods. Forty-four full text manuscripts were reviewed, addressing the incidence, natural history, presenting symptoms, surgical management, postoperative course, and associated syndromes for osteomas of the facial skeleton. Twenty-six of these presented cases of osteomas specifically involving the orbit (Table 2). Most orbital osteomas occurred in patients in their fourth and fifth decades. Surgical approaches described include endoscopic, coronal, transcaruncular, transblepharoplasty incision, and combined. Over the last 5 years, most surgical cases reported in the literature were completely excised endoscopically. Those that were incompletely resected typically underwent a second procedure to remove residual tumor. Recurrence was extremely rare, with only 1 study reporting recurrent frontal sinus osteoma 7 years after initial surgery. Selected Cases. Case 1. This case highlights subtotal resection as a safe and effective management option for orbital osteomas. A 53-year-old man
presented with a 6-month history of left hyperglobus and mass effect on the inferior orbital rim. Best corrected visual acuity (BCVA) was 20/25, and the remainder of the examination was normal with the exception of 1 mm left exophthalmos and a palpable bony mass of the left inferior orbital rim (Fig. 1). Imaging showed a 2.2 × 2.1 × 1.6 cm osteoma of the left orbital floor abutting the left globe inferiorly and displacing the orbital soft tissues superiorly. The patient underwent planned near-total resection of the lesion by sculpting the osteoma flush with normal contour of the floor and inferior orbital rim via a lower eyelid transconjunctival approach with lateral canthotomy and cantholysis. Histopathology showed a composite of woven and lamellar bone, deposited in a cortical pattern with Haversian-like systems with scant fibrous stroma. The portion of the osteoma involving the zygoma outside the orbit was left intact. On follow-up month 3, the patient’s eye was in normal position, motility was intact, and he was orthophoric, with BCVA 20/15. Case 2. This case demonstrates the potentially protean nature of sinoorbital osteomas and the possible systemic association with Gardner syndrome. A 15-year-old woman presented with progressively worsening blurry vision and metamorphopsia of the left eye for 1 year. She had also developed headaches and a pressure sensation behind her left eye over several months prior to presentation. On examination, BCVA OS
TABLE 2. Summary of selected osteoma case reports and series with emphasis on the years 2007–2012 Study
Year
n
2012
1
15
F
Frontal sinus
External, multistage
None
Kayaci et al.8 Koktekir et al.11 Nicolotti et al.31
2012 2012 2012
1 1 1
80 16 62
F M M
Sphenoid Ethmoid Frontal sinus
None None None
Pons et al.5
2012
25
44
Turri-Zanoni et al.28 2012
60
44.1
Yazici et al.23
2012
3
18
Coronal Endoscopic Direct frontal excision w/titanium 4 coronal, 19 endoscopic, 2 combined 4 external, 31 endo, 25 combined External
Friedrich9
2009
1
53
Livaoĝlu et al.29
2009
1
55
Miman et al.38
2009
1
28
Seiberling et al.16
2009
23
47
Kashkouli et al.7 Gerbrandy et al.39 Andaloussi et al.40
2008 2007 2006
1 1 3
22 39 26.3
Selva et al.1 Sibony et al.41 Eppley et al.42 Ma’luf et al.43 Naraghi et al.44 Selva et al.15 Becelli et al.6 Sires et al.10 Alper et al.45 Kobayashi et al.46 Naidu et al.47
2004 2004 2003 2003 2003 2003 2002 1999 1998 1996 1987
11 1 2 1 1 1 2 1 1 2 1
Ns 54 39.5 27 42 28 39.5 15 22 2 15
Albert et al.48
1982
1
Fan et al.
12
Avg age
74
Gender
Osteoma origin
Surgical technique
16 M, 9 F Ethmoid sinus (24% Orbit) 33 M, 27 F Frontal/ethmoid (10% Orbit) 3M Ethmoid/ frontoethmoid F Primary Orbit (Floor) Infraorbital with Ethisorb
Complications
None
Outcome Continued reconstruction of lower face Sx improved Sx resolved Sx resolved, forehead numbness No recurrence
Persistent Complete resection in diplopia (1) all but 2 cases None Sx resolved, no recurrence None Resolved sx and no recurrence M Primary orbit (roof) Blepharoplasty incision None Resolved sx and no recurrence F Ethmoid sinus Endoscopic None Sx resolved, mild enophthalmos 11 M, 12 F Frontal sinus (4.3% 13 endo, 10 external, Frontal sinus Sx resolved, no Orbit) 2 combined narrowing (1) recurrence M Primary orbit (roof) Coronal None Sx resolved F Ethmoid Combined None Sx resolved 3F Frontal/ethmoid External Ptosis and Sx resolved diplopia (1) Ns Frontal/ethmoid Ns (only 9 required surgery) Ns 1 recurrence F Ethmoid External None Sx resolved 1 M, 1 F Frontal sinus External None Sx resolved M Ethmoid Combined None Sx resolved M Ethmoid Endoscopic None Sx resolved F Frontal/ethmoid Endoscopic and eyelid crease None Sx resolved 1 M, 1 F Primary orbit Coronal, subciliary None Complete excision M Ns Blepharoplasty incision None Sx resolved M Ethmoid Coronal None Sz resolved 1 M, 1 F Frontal/maxillary Coronal None Sx resolved M Primary orbit (medial Coronal None Sx resolved wall) F Ethmoid/maxillary External None Sx resolved
Ns, not specified; endo, endoscopic; sx, symptoms. A series was included only if it contained orbital cases.
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FIG. 1. Top Left: Case 1, 53-year-old man with left hyperglobus and palpable inferior rim deformity due to a sino-orbital osteoma. Top Right: Axial CT showing left maxillary sinus/orbital floor osteoma. Bottom Left: Intraoperative photographs after partial resection and sculpting of the floor and inferior rim. Bottom right: Hematoxylin-eosin stain of specimen. Note composition of mostly mature-appearing lamellar bone with Haversian-like systems. was 20/80 with no motility deficits or afferent pupillary defect. She had 2 mm of left telecanthus and 2 mm of proptosis (Fig. 2). Funduscopic examination revealed choroidal folds in the macula and indentation of the medial aspect of the globe. A large left ethmoidal osteoma impinging on the medial rectus and small right ethmoidal osteoma were found on imaging. The patient underwent image-guided transnasal endoscopic resection of both osteomas. At follow-up month 4, the patient’s BCVA OS was 20/40, and she reported no further headaches or eye pain. Her left fovea returned to its normal contour with resolution of the choroidal folds, although she continued to have a mild epiretinal membrane. A small amount of residual tumor was left at the attachment site along the left frontal recess and cribriform plate. The patient was also referred for genetic evaluation for Gardner syndrome, as her father had a history of multiple colon polyps. At 1-year follow up, the patient was asymptomatic and did not show any growth of the residual osteoma on nasal endoscopy or CT scan. Neither she nor her family members have undergone further work-up for Gardner syndrome. Case 3. This case demonstrates the importance of a multidisciplinary surgical approach to large sino-orbital osteomas. A 17-year-old man presented with severe right inferior globe displacement and 12 mm of right proptosis (Fig. 3). On imaging, he was found to have a large right frontal sinus osteoma with secondary extension in the orbit. The patient underwent complete resection via coronal approach with an osteoplastic flap. The anterior table of the frontal sinuses was noted to be thin during the initial resection, and the frontal sinus was obliterated using a temporalis/galea flap to increase the chance of survival of the anterior table. The orbital roof was reconstructed with porous polyethylene titanium mesh (Stryker, Kalamazoo, MI, U.S.A.). No complications were noted at 4-month follow up, and the patient had resolution of proptosis and no new visual complaints. Case 4. This case also demonstrates the viability of partial resection as a treatment option in selected patients. An 18-year-old woman presented with eye pain and a right medial orbital palpable nodule for 1 year. On examination, BCVA was 20/25 in the affected eye, and she had a superomedial nonmobile mass with 1 mm proptosis. She was found to have a 2.6 × 2.2 × 1.8 cm right ethmoid osteoma extending in the right orbit, bulging up to the lateral lamella of the cribiform plate and causing frontal recess obstruction. She underwent partial resection/drilling
of the orbital component of the osteoma via transcaruncular approach to relieve symptoms from mass effect. The ethmoidal component of the osteoma was initially left undisturbed, given its proximity to the cribriform plate. However, she continued to have frontal recess obstruction and underwent a second surgery for complete excision via a modified Lynch incision. At the time of excision, she also underwent medial wall reconstruction with a porous polyethylene implant. Pathologic study of the resected bone fragments showed anastomosing trabeculae of woven bone and spaces filled with vascular loose connective tissue. At 2-month follow up, her proptosis was completely resolved, and symptoms were improved. She had mild diplopia in upgaze only.
DISCUSSION Sino-orbital osteomas are found incidentally on 1% of plain sinus radiographs and 3% of CT scans and are often small and asymptomatic.3,4,18 Given their slow growth rate (average 1.6 mm/year), asymptomatic or incidentally discovered lesions are typically observed.19 Approximately 5% of osteomas become symptomatic or require surgery.4 Osteomas may be more common in men, with the reported male to female ratio ranging from 1:1 to 2.4:1,2 although this predilection was not seen in this series. The differential diagnosis for orbital osteoma includes benign entities such as ossifying fibroma, fibrous dysplasia, osteoblastoma, and malignant neoplasms such as osteosarcoma and osteoblastic metastases.1 In the head and neck, osteomas are most commonly found in the paranasal sinuses—classically the frontal sinus—although some case series have found ethmoid osteomas to be more prevalent.2,20 Ethmoid sinus osteomas often originate from the lateral portion of ethmoid sinus roof and extend toward the orbit and also toward the frontal sinus.5 Grossly, sino-orbital osteomas have a whitish coloration and a smooth or bosselated surface. Histologically, osteomas exhibit variable patterns, most commonly mature lamellar bone with trabeculae.4 Several classification schemes based on histology and morphology have been used to describe osteomas. One common scheme differentiates lesions in ivory (compact), mature (cancellous), and mixed ivory/mature subtypes based on histology.21
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FIG. 2. A, Case 2, 18-year-old woman with left telecanthus, proptosis, and lateral globe displacement from an ethmoid osteoma. B, Fundus photograph showing macular choroidal folds. C, Coronal CT showing large left ethmoidal osteoma displacing the globe laterally and causing proptosis. Note the thick calcified rim and polypoid configuration. D, Hematoxylin-eosin stain of cortical area of specimen. E, Hematoxylin-eosin stain of trabecular area of specimen. F, Medium magnification of central osteoblastoma-like area of specimen.
Others suggest this classification scheme has little clinical relevance and instead describe lesions morphologically based on grossly organized concentric zones of bone maturation.1 The “osteoma with osteoblastoma-like features” histologic subtype is marked by interanastomosing trabeculae and fibrovascular stroma surrounded by osteoblasts and osteoclasts.22,23 Osteomas with osteoblastoma-like features are more likely to exhibit extracavitary extension, cause visual symptoms, and be incompletely excised; however, they may be less likely to recur than other osteomas.22 They also have a distinct radiographic zonal appearance, with lower density osteoblastoma-like material at the core and denser mature bone in the periphery.23 On imaging, osteomas appear often as dense, sclerotic, well-defined masses forming a polypoid configuration. CT is the imaging modality of choice, given its excellent rendering of bony structures.23 MRI may give a falsely aggressive appearance to osteoid osteoma. More accurate radiologic
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diagnoses are made by CT as opposed to MRI.24 However, in the case of possible intracranial extension, MRI is also recommended.25 Indications for surgical intervention include vision loss or optic neuropathy, eye pain, proptosis or globe displacement, diplopia, palpable defect, postobstructive sinus disease, chronic headaches, or cosmetic deformity.4,26 Georgalas et al.27 proposed treatment when the following criteria are met: 1) Osteomas associated with symptoms after all other explanations for the symptoms have been excluded, 2) Large (extending to >50% of the frontal sinus) or growing osteomas, as seen on serial CTs, and 3) Osteomas associated with current complications (mucocele, orbital symptoms, neurologic symptoms, external deformity) or imminent (complete obstruction of the frontal recess or intraorbital/intracranial extension) complications. Because growth appears to stem outward from a central core, partial resection that includes removal of this core is also regarded to be a viable alternative to complete resection.28 Several surgical approaches to primary and secondary orbital osteomas have been described.28–30 A transconjunctival inferior fornix approach is useful for inferior orbital lesions. Anterior orbitotomy by lateral canthotomy with swinging lower eyelid approach provides exposure for inferior anterior orbital lesions. Superior lesions may be accessed via an upper eyelid crease incision, but extensive lesions may also require the wider exposure afforded by a coronal incision. The coronal approach is useful for giant frontal osteomas (see Case 3), requiring reconstruction for optimal functional and cosmetic results.31 A transcaruncular or medial canthal cutaneous approach, with or without nasal endoscopy, may be suitable for superomedial osteomas. The endoscopic approach is preferred for many ethmoid osteomas and can be used judiciously for frontal sinus osteomas and even selected osteomas with intraorbital extension.5,25,28 Aspects to consider for endoscopic removal of frontal sinus osteomas include 1) location of base of attachment, 2) location in relation to a virtual sagittal plane through the lamina papyracea, and 3) relative size of lesion in comparison with the frontal recess.26 Many frontal sinus osteomas can be removed endoscopically with the exception of those with a superior pedicle, far lateral location, or large size compared with the frontal recess.16,26,32 For complete resection of osteomas, greater surgical exposure is necessary than for resection of similarly sized soft-tissue tumors. Because reconstructions depend on intraoperative interpretation of the bony defect, and the natural and highly variable topography of the orbit, no single set of criteria (for defect size or location) is applicable. The choice of a particular approach depends not only on the size and location of the osteoma but also the experience of the surgeon. Autografting, alloplastic implantation, and sculpting of the osteomatous bone represent effective reconstruction techniques. Although developmental abnormality, trauma, and infection/chronic inflammation have been proposed as possible mechanisms, the etiology of osteomas remains unknown.1,6,14 Most osteomas are sporadic. However, they may be associated with Gardner syndrome (phenotypic variant of Familial Adenomatosis Polyposis or FAP), an inherited colonic adenomatosis in which the incidence of colon adenocarcinoma is 100%. Other manifestations of Gardner syndrome include extracolonic malignancies such as papillary thyroid carcinoma, mesenteric fibromatosis, hepatoblastoma, and gastric carcinoma.33,34 Gardner syndrome is also characterized by benign extracolonic lesions such as nasal angiofibromas, adrenal adenomas, and congenital hyperplasia of the retinal pigment
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
Orbital Osteoma
FIG. 3. A, Case 3, 17-year-old man with severe right proptosis and inferior globe displacement from frontal sinus osteoma. B, Coronal CT showing large frontal sinus osteoma with orbital extension and globe displacement. C, Postoperative photograph showing improvement in eye position and reduced proptosis. D, Intraoperative photograph showing bicoronal flap and unroofed frontal sinus. E, Intraoperative photograph showing completely resected right frontal sinus osteoma. F, Intraoperative photograph after obliteration of the frontal sinus with temporalis/galea flap and porous polyethylene titanium mesh roof reconstruction.
epithelium (CHRPE), which are usually multiple and bilateral.34,35 On average, clinically evident osteomas are found 17 years before a diagnosis of Gardner syndrome is made.36 As both the colonic and extracolonic manifestations of this syndrome are life threatening, often at a young age, the oculoplastic surgeon can impact outcome by initiating a work-up for early detection. Patients with osteoma and suspicion of Gardner syndrome (positive family history, gastrointestinal symptoms, and/or extracolonic manifestations) should have a dilated funduscopic exam, genetics consult with possible testing for adenomatous polyposis coli mutations, and Gastroenterology consult for possible flexible sigmoidoscopy or colonoscopy.36,37 While this series contains several paranasal sinus osteomas with orbital extension, it is limited by relatively few cases of primary orbital osteoma—a function of this lesion’s extremely low prevalence. Follow-up time (mean 18 months) was also limited in many of these cases. It is possible that additional morbidity related to wide/extensive excision or recurrence may be discovered with greater follow up. Given the slow general growth and benign nature of these tumors, subtotal resection is reasonable when complete excision would create disproportionate morbidity or significantly complicate surgery or reconstruction. Patients should be monitored for recurrence or growth of the residual tumor with annual clinical exams and biannual imaging. If clinical and radiographic stability is reached after a period of 5 years, imaging can be performed on an as-needed basis for new symptoms or examination findings. Drawing from 4 separate academic institutions, this study was unable to control for variations in clinical recording, surgical technique, histologic review, and follow up among senior
authors. Also, the geographically and culturally diverse populations contributing these cases make discerning common etiologic risk factors difficult.
CONCLUSIONS Although osteomas are the most common bony tumor of the paranasal sinuses, orbital extension and primary orbital osteoma are rare and can present variably. Radiographic, gross, and histologic variations appear to have little impact on clinical behavior. A multidisciplinary surgical team may be required, potentially using both endoscopic and open approaches. Surgical goals may include partial or complete resection, in addition to reconstruction by bony sculpting and/or implantation of alloplastic implant materials. Partial resection is a viable and potentially preferable alternative when complete resection risks excessive morbidity or unnecessary surgical complexity. Overall, clinical outcomes of symptomatic lesions are favorable.
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