Journal of Clinical Neuroscience 21 (2014) 2233–2238
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Technical Note
The modified hemi-Lothrop procedure: A variation of the endoscopic endonasal approach for resection of a supraorbital psammomatoid ossifying fibroma James K. Liu a,b,c,⇑, Zachary S. Mendelson a, Pariket M. Dubal b, Neena Mirani d, Jean Anderson Eloy a,b,c a
Department of Neurological Surgery, Rutgers New Jersey Medical School, 90 Bergen Street, Suite 8100, Newark, NJ 07103, USA Department of Otolaryngology, Head & Neck Surgery, Rutgers New Jersey Medical School, Newark, NJ, USA Center for Skull Base and Pituitary Surgery, Neurological Institute of New Jersey, Rutgers New Jersey Medical School, Newark, NJ, USA d Department of Pathology and Laboratory Medicine, Rutgers New Jersey Medical School, Newark, NJ, USA b c
a r t i c l e
i n f o
Article history: Received 15 July 2014 Accepted 28 July 2014
Keywords: Endoscopic endonasal approach Endoscopic skull base surgery Juvenile psammomatoid ossifying fibroma Modified hemi-Lothrop Skull base tumor Supraorbital tumor
a b s t r a c t Tumors in the supraorbital region are most commonly accessed through transcranial approaches, including fronto-orbital, orbitozygomatic, and eyebrow supraorbital keyhole approaches. Purely endoscopic endonasal approaches (EEA) are more challenging to perform because of limitations in access and visualization for lateral extension beyond the midline corridor. The modified hemi-Lothrop procedure, a variation of an extended EEA, allows for binostril access and visualization of the lateral supraorbital region while preserving the contralateral frontal sinus drainage pathway. The operative technique and nuances are illustrated in a rare case of a supraorbital juvenile psammomatoid ossifying fibroma (JPOF) causing symptomatic orbital compression. The key components of the approach consisted of an endoscopic Draf IIB (left frontal sinusotomy) ipsilateral to the tumor, and a superior septectomy for binostril bimanual instrumentation. Excellent visualization, access, and tumor removal of the supraorbital region was achieved with angled endoscopy and curved instrumentation from the contralateral nasal cavity and through the septectomy window (‘‘cross-court’’ trajectory). The modified hemi-Lothrop procedure with angled endoscopy is a safe and effective alternative route to traditional transcranial approaches to access the supraorbital region. To our knowledge, this is the first case of a supraorbital JPOF that was successfully resected via a purely EEA. Ó 2014 Elsevier Ltd. All rights reserved.
1. Introduction Ossifying fibromas are benign, locally invasive fibro-osseous lesions with a predilection for the bones of the craniofacial skeleton [1]. Ossifying fibroma is a broad term that describes a spectrum of skull lesions [2], a subset of which is juvenile ossifying fibroma (JOF). One type of JOF is juvenile psammomatoid ossifying fibroma (JPOF) [2]. JPOF is most common in younger age groups and is characterized by a variable fibroblastic stroma containing calcified spherical masses of osteoid that have the appearance of a psammoma body [2,3]. JPOF typically arises in the paranasal sinuses, orbit, and the fronto-ethmoid complex [4]. Extension into the orbit and anterior skull base has also been observed [3]. Due to its aggressive nature, a complete surgical excision is recommended in order to reduce the risk of recurrence [3,5]. The open surgical approach has been the standard for the surgical management of JPOF, including transfrontal-nasoorbital, ⇑ Corresponding author. Tel.: +1 973 972 2906; fax: +1 973 972 2333. E-mail address:
[email protected] (J.K. Liu). http://dx.doi.org/10.1016/j.jocn.2014.07.012 0967-5868/Ó 2014 Elsevier Ltd. All rights reserved.
transfrontal-nasal, combined subfrontal and nasal, and craniofacial approaches [3,6]. We report a successful resection of a supraorbital psammomatoid ossifying fibroma using an endoscopic endonasal approach to the supraorbital region, namely the modified hemi-Lothrop procedure. This approach uses binostril access with a superior septectomy to allow bimanual maneuverability to target lesions of the supraorbital region, including the supraorbital frontal sinus and supraorbital ethmoid cell [7–9]. We describe the surgical technique and operative nuances of this approach as illustrated in this patient with a supraorbital ossifying fibroma causing symptomatic orbital compression. This study was approved by the Institutional Review Board. 2. Operative technique and case illustration 2.1. Clinical presentation The patient presented with progressive headaches and intermittent blurry vision in the left eye. The patient also reported nasal congestion and hyposmia over a 2 year period. An MRI and
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subsequent CT scan demonstrated an expansile supraorbital mass extending from the left suprabullar ethmoid cells into the left supraorbital ethmoid cells, with extension into the skull base and compression of the orbital roof (Fig. 1, 2). There was no evidence of intracranial extension.
2.2. Surgical approach and operative pearls The patient underwent an endoscopic endonasal approach via a modified hemi-Lothrop procedure for resection of the left supraorbital mass (Fig. 3, 4). In summary, the procedure consisted of a left anterior middle turbinectomy, left total ethmoidectomy, and antrostomy into the left maxillary sinus. An endoscopic Draf IIB (left frontal sinusotomy) with a superior septectomy was performed to allow binostril and bimanual placement of instrumentation and angled endoscopes to access and visualize the lateral supraorbital recess from the contralateral nasal cavity (Fig. 3D). The patient was positioned supine with his head placed in three-pin skull fixation. The head was extended slightly to facilitate access to the anterior skull base and frontal sinus region. The nose and nares were prepped with Betadine (Sanofi, Paris, France) followed by Afrin-soaked pledgets (Merck, Whitehouse Station, NJ, USA).
The nasal cavity was examined bilaterally with a 4 mm 30 degree rigid endoscope. The left uncinate process, axilla of the middle turbinate, and the head of the middle turbinate were injected with 1% lidocaine with 1:100,000 epinephrine. Initially working through the left nostril with a 30 degree endoscope, a left maxillary antrostomy, total ethmoidectomy, and sphenoid sinusotomy were performed for adequate exposure. At this juncture, the mass was clearly visualized extending from the suprabullar ethmoid cells into the supraorbital ethmoid cells and extending along the fovea ethmoidalis (skull base). Multiple segments of the left lamina papyracea were then removed with a combination of Blakesley forceps and a frontal sinus curette with preservation of the periorbita. Next, a left Draf IIB frontal sinusotomy (opening of the frontal sinus recess and floor of the frontal sinus from the lamina papyracea laterally to the nasal septum medially) was performed. A superior septectomy was then performed by removing the portion of the quadrangular cartilage and perpendicular plate of the ethmoid just inferior and anterior to the frontal sinus opening. This maneuver created a working corridor to pass a 30 degree and 70 degree endoscope and angled instrumentation from the right nostril to visualize the left supraorbital ethmoid and left lateral frontal sinus recess (Fig. 3D, 4).
Fig. 1. Preoperative MRI (A: axial T2-weighted; B: coronal T2-weighted; C: sagittal T1-weighted post-gadolinium; and D: coronal T1-weighted post-gadolinium) demonstrates a homogenous enhancing supraorbital mass (asterisk) arising from the left supraorbital ethmoid air cell invading the superomedial orbit with mass effect and compression on the orbital contents. The mass extends to the anterior skull base and superior orbital rim. There is mucosal thickening of the left frontal sinus consistent with sinusitis.
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Fig. 2. CT scan (A, B: coronal views; C: axial view; and D: sagittal view) demonstrates an expansile lesion (asterisk) within the left supraorbital ethmoid sinus with ground glass density, exerting mass effect on the superomedial orbit. The drainage of the lateral supraorbital ethmoid cell is obstructed by the tumor (arrow).
The lesion was entirely extradural and well-encapsulated, containing calcified material. Several biopsies were taken, which came back as ossifying fibroma. A medial orbital wall decompression was performed via removal of the lamina papyracea. This allowed for the displacement of the periorbita laterally for access to the supraorbital roof, which incorporated the inferior wall of the lesion. The margins of the tumor were then drilled off starting with the inferior aspect and going out laterally. An angled frontal sinus curette was used to help dissect the tumor in an extracapsular fashion (Fig. 4). Finally, a high-speed drill was used to ‘‘polish’’ the surrounding bone of the resection bed until normal appearing bone was visualized. A gross total resection of the tumor was achieved. The surrounding bone was inspected and there appeared to be no further tumor in the resection bed. Angled 30 degree and 70 degree endoscopes were then used to inspect the cavity and a partition between the supraorbital ethmoid cell and frontal sinus was resected, creating a single supraorbital ethmoid/frontal sinus cavity. There was no evidence of intraoperative cerebrospinal fluid leakage. The superior and medial walls of the orbit were sufficiently decompressed. 2.3. Histopathologic examination Histopathologic examination demonstrated a well demarcated proliferation of mature lamellar bone admixed with fibrous stroma at the periphery and uniform small ossicles (psammomatoid
bodies) embedded in a relatively cellular spindle cell stroma in the central portion of the lesion. These findings were consistent with an ossifying fibroma, psammomatoid type (Fig. 5). 2.4. Postoperative course Postoperatively, the patient was neurologically intact with no evidence of visual disturbance. Postoperative MRI demonstrated complete resection of the tumor without any evidence of residual tumor (Fig. 6). The left orbit was well decompressed. The remainder of the hospital course was uneventful and the patient was discharged on postoperative day 2. There was no evidence of postoperative cerebrospinal fluid leak. The patient remained free of recurrence at 3 years postoperatively. 3. Discussion 3.1. JPOF The mainstay of therapy of JPOF is complete surgical resection with 5 mm margins [10]. Recurrence is very likely when incompletely resected, with rates in the literature ranging from 30–56% [11,12]. Open surgical procedures, such as transcranial, transorbital, and transfacial approaches, may require craniofacial and orbital reconstruction, depending on the degree of expansion and location of the lesion [13]. One of the major advantages of the endoscopic endonasal approach is the avoidance of facial incisions and
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Fig. 3. (A) Illustration of the tumor in the coronal plane demonstrating the dissection technique for the modified hemi-Lothrop procedure. Via the ipsilateral left nasal cavity, a Draf IIB is performed (dotted arrow). An anterior superior septectomy is performed and the left supraorbital tumor is accessed via the contralateral nasal cavity (solid arrow). (B) Coronal CT scan after a modified hemi-Lothrop procedure demonstrates complete removal with excellent orbital decompression. (C) A 30 degree angled endoscope placed in the left nostril offers limited view of the lateral supraorbital region. (D) A 30 degree angled endoscope is placed in the contralateral nasal cavity via the right nostril which provides excellent visualization and access of the tumor in the lateral supraorbital region. Ó 2014 Chris Gralapp, reprinted with permission (This figure is available in colour at http://www.sciencedirect.com/).
Fig. 4. (A–C) Intraoperative endoscopic view of the tumor removal from the left lateral supraorbital ethmoid using a 30 degree angled endoscope placed in the right nasal cavity through the anterior superior septectomy defect. The tumor (asterisk) is visualized in the left supraorbital ethmoid cell and removed with angled suctions, curettes, and rongeurs. The orbital roof and medial orbital wall is decompressed with a high-speed diamond drill and curettes with preservation of the periorbita (PO). (D) Final view of the resection bed shows the fovea ethmoidalis (FE) of the anterior skull base and the frontal sinus recess (FSR, dotted circle) which is located more anterior and lateral to the FE (This figure is available in colour at http://www.sciencedirect.com/).
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Fig. 5. Histopathologic examination (A, hematoxylin and eosin, original magnification 100) revealed well proliferation of mature lamellar bone admixed with fibrous stroma at the periphery of the lesion. At higher magnification (B, hematoxylin and eosin, original magnification 400), small ossicles (psammomatoid bodies) embedded in a relatively cellular stroma composed of spindle cells in the central portion of the lesion are visualized, most consistent with a juvenile ossifying fibroma (This figure is available in colour at http://www.sciencedirect.com/).
Fig. 6. Postoperative MRI (A: coronal T1-weighted post-gadolinium; and B: axial T2-weighted) demonstrates complete resection of the tumor without evidence of residual disease. The pathway of the modified hemi-Lothrop from the right nasal cavity through the superior septectomy corridor to the left supraorbital region is demonstrated by the white arrow (A).
craniotomy while preserving the integrity of the craniofacial skeleton and overlaying structures. The location of this tumor was primarily in the left supraorbital ethmoid air cell which made it favorable for an endoscopic endonasal approach via a modified hemi-Lothrop procedure [7,8]. The tumor appeared to arise from the supraorbital ethmoid cell, which is an accessory ethmoid cell in the frontal region that extends into and pneumatizes superolaterally along the orbital plate of the frontal bone. This cell is located posterolateral to the frontal sinus and drains posterolaterally to the frontal sinus recess. There is a common wall that divides the frontal sinus from the supraorbital ethmoid cell, which is often mistaken as the lateral extension of the frontal sinus. The vector of globe displacement from lesions arising in the supraorbital ethmoid cell is typically oriented inferiorly, as demonstrated in the present case. This is in contrast to frontal sinus lesions that typically displace the orbital contents inferolaterally, and ethmoid lesions that tend to displace the orbit laterally [14]. 3.2. Modified hemi-Lothrop procedure The modified hemi-Lothrop procedure has been demonstrated previously in the endoscopic access to lateral supraorbital frontal
sinus lesions, as well as in the drainage of mucoceles of the supraorbital ethmoid cell [9,14,15]. Although this technique is well suited to treat sinus disorders, it is not well known in the neurosurgical literature to treat neoplastic skull base lesions. The key maneuvers of this approach include an ipsilateral endoscopic Draf IIB frontal sinusotomy and a superior septectomy. The superior septectomy window allows binostril access and bimanual instrumentation. The endoscope can be placed in either the ipsilateral or contralateral nostrils. The endoscopic view through the ipsilateral nostril is limited and inadequate when attempting to visualize far lateral lesions of the frontal sinus and supraorbital ethmoid cells, even with the use of angled endoscopes. On the other hand, placement of angled 30 degree and 70 degree endoscopes into the contralateral nostril and through the septectomy window allows for extreme lateral (‘‘cross-court’’) visualization of this supraorbital region (Fig. 3). In addition to angled endoscopy, angled and curved instrumentation (drills, curettes, rongeurs and suctions) are critical for successful tumor dissection in the supraorbital region when using this approach. Therefore, the surgical pearl in this technique is to perform an adequate superior septectomy corridor to allow access and visualization via the contralateral nostril without having to
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violate the contralateral frontal sinus recess. Only unilateral opening of the ipsilateral frontal sinus is required, and a full modified Lothrop procedure (bilateral frontal sinusotomy or endoscopic Draf type III) is avoided in order to preserve the mucociliary drainage pathway of the nondiseased contralateral frontal sinus. In the present case, these advantages were crucial, as JPOF must be removed completely to prevent tumor recurrence. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References [1] Makek MS. So called ‘‘fibro-osseous lesions’’ of tumorous origin. Biology confronts terminology. J Craniomaxillofac Surg 1987;15:154–67. [2] El-Mofty S. Psammomatoid and trabecular juvenile ossifying fibroma of the craniofacial skeleton: two distinct clinicopathologic entities. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:296–304. [3] Sarode SC, Sarode GS, Waknis P, et al. Juvenile psammomatoid ossifying fibroma: a review. Oral Oncol 2011;47:1110–6. [4] Han MH, Chang KH, Lee CH, et al. Sinonasal psammomatoid ossifying fibromas: CT and MR manifestations. AJNR Am J Neuroradiol 1991;12:25–30.
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