CRANIOMAXILLOFACIAL DEFORMITIES/COSMETIC SURGERY

Correction of a Contour Deformity Associated With Frontal Pneumosinus Dilatans Using Surgical Navigation Technology Joseph A. Ricci, MD,* Naman S. Desai, MD,y and Nicholas Vendemia, MDz Pneumosinus dilatans (PD) is a pathologic hyperaeration of the paranasal sinuses of unknown etiology. Although benign itself, PD has been associated with a number of serious concomitant conditions, including meningioma, optic nerve tumors, and visual loss. Patients with PD often present with cosmetic complaints, desiring recontouring of the facial bones to achieve an improved appearance of the face. The present case illustrates one of the first attempts at intraoperative surgical navigation to map the frontal sinus during correction of the facial deformity caused by PD. The navigation device was used to give the surgical team real-time information during the case to prevent violation of the posterior table of the frontal sinus, allowing for facial bone contouring to occur in a more efficient and safer manner by way of accurate osteotomy placement with no wasted bone for reconstruction and no accidental intracranial involvement. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 73:529-533, 2015 Pneumosinus dilatans (PD) is a pathologic condition of unknown etiology, causing hyperaeration of 1 or more of the paranasal sinuses, leading to a significant facial deformity. The entity was first described by Meyers in 1898, although the term ‘‘pneumosinus dilatans’’ was not coined until 1918 by Benjamin.1 The frontal sinus is the most common paranasal sinus involved, with PD presenting on average between ages 20 and 40 years.2 An unsightly, Cro-Magnon–like brow has been the most common presenting complaint for patients with PD. Although patients present most often with aesthetic complaints, numerous serious associated conditions have been reported with PD, including intracranial tumors, arachnoid cysts, nasal obstruction, mucoceles, and orbital tumors, leading to exophthalmos and blindness.3 The rate of associated conditions has been greater when multiple sinuses are involved.2 Although the etiology of PD is unknown, several theories have been proposed as the cause of sinus

hyperaeration, including obstruction of the frontonasal duct with a ball valve-like mechanism, gas forming bacteria, fibrous dysplasia, and hormonal dysregulation.1,4,5 In the present study, we report the case of 33-year-old man with frontal sinus PD who had presented with progressive enlargement of his brow, which was corrected with the assistance of a real-time intraoperative navigation system to prevent untoward entry into the cranial vault.

*Resident, Division of Plastic Surgery, Department of Surgery,

General Hospital, 55 Fruit St, WACC 435, Boston, MA 02114;

Case Report The patient was a 33-year-old man who presented with a complaint of progressive enlargement of his forehead and brow that made him appear ‘‘angry and unhappy’’ to others. His appearance bothered him to the point that he wore a hat at all times when in public and had styled his hair to hide his brow. The patient reported that his brow had begun enlarging in childhood, had slowed as he entered adulthood, and then

Massachusetts General Hospital, Harvard Medical School, Boston, MA.

e-mail: [email protected] Received July 19 2014

yResident, Department of Radiology, Brigham and Women’s

Accepted August 18 2014 Ó 2015 American Association of Oral and Maxillofacial Surgeons

Hospital, Harvard Medical School, Boston, MA. zAttending Surgeon, Manhattan Aesthetic Surgery, New York, NY.

0278-2391/14/01372-X

Address correspondence and reprint requests to Dr Ricci:

http://dx.doi.org/10.1016/j.joms.2014.08.023

Division of Plastic Surgery, Department of Surgery, Massachusetts

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530 began to grow again rapidly 2 years before presentation. He reported headaches and the occasional sensation of pressure in his forehead, but denied any other symptoms. The findings from the physical examination were consistent with the patient’s complaints in that his brow was a defining feature in the appearance of his face, with significant bossing of his entire supraorbital brow (Fig 1). The anterior rhinoscopic examination findings were unremarkable, and maxillofacial computed tomography (CT) revealed hyperaeration of both of his frontal sinuses beyond the normal anatomic boundaries. However, the bony walls of the frontal sinuses were normal in caliber, and the remaining paranasal sinuses were normal in appearance (Fig 2). The CT scan confirmed the suspected diagnosis of frontal sinus PD. The patient was taken to the operating room for recontouring of his brow and obliteration of the frontal sinus. After induction of anesthesia, the surgical navigation equipment, BrainLAB (BrainLAB, Inc, Westchester, IL), was attached. BrainLAB provides real-time cross-sectional images of the craniofacial skeleton by automatically triangulating dozens of separate anatomic points delineated by the combination of hardware attached to the skull, by sterile navigational instruments used intraoperatively, and by the BrainLAB machine itself (Fig 3). At the start of the procedure, preoperative CT imaging data were transferred to the BrainLAB navigation

SURGICAL NAVIGATION TO TREAT PNEUMOSINUS DILATANS

system. Using this information, the borders of the frontal sinus were identified and marked within the planning software. The tracking system consists of 2 infrared cameras that track the spatial position and orientation of markers, which emit infrared light by light-emitting diodes. Intraoperatively, a rigid marker is attached to the patient’s body, with bone screws directly to the skull. A second marker is attached to a sterile and freely mobile pointing device. The navigation system processes the tracking information by seeing the tip of the pointer continuously on the screen of the navigation system with a triplanar view of the image data and a 3-dimensional model of the patient’s anatomy. Once in place, the BrainLAB system assisted the team with the identification and exposure of the anterior table of the frontal sinus after a standard bicoronal incision. Once the frontal bones and supraorbital rims were exposed, the BrainLAB instrumentation was used to define the borders of the hyperaerated frontal sinuses in real time using the sterile surgical navigation probe. This allowed for very accurate and safe placement of the anterior table osteotomies, preventing unwanted violation of the cranial vault. The entire deformed segment of the frontal bone was removed en bloc, exposing the frontal sinus cavities, which were empty (Fig 4). The mucosa was removed using a pneumatic burr, and samples were sent for pathologic analysis. No obvious pathologic features were

FIGURE 1. Preoperative photographs of the patient in the A, frontal and B, lateral views. Note the bossing of the brow, particularly evident in the lateral view and the patient’s hairstyle, designed to camouflage his enlarged brow. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

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obtained, the osteotomized segment of frontal bone was plated to the intact skull (Fig 5). The soft tissue of the forehead was redraped, and the incision was closed in the standard fashion. Of note, once the skin was redraped, the plates were not visible or palpable in our patient. However, in a thinner skinned individual, these plates might have been substituted with a 0.5-mm mesh and 1.0-mm or 1.3-mm screws or bioresorbable hardware. The aesthetic outcome was excellent at the patient’s 6-month follow-up visit, and he expressed complete satisfaction with the procedure. He no longer felt insecure about the appearance of his brow in public situations and no longer felt the need to wear a hat to leave his house (Fig 6). FIGURE 2. Preoperative computed tomography scan showing hyperaeration of the frontal sinuses. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

evident on inspection of the sinus cavities. After mucosal obliteration, the frontal sinus ostia were filled with deep temporal fascia, and the sinus cavities were filled with subcutaneous abdominal fat. The excised segment of frontal bone was osteotomized at several key points to allow for a flatter, more uniform, contour. The bony segments were then plated together using 2.0-mm titanium miniplates, and the new anterior surface of the bone was refined with a pneumatic burr. The edges of the osteotomized segment and the remaining frontal bone were carefully contoured obliquely to allow for insetting of the entire segment into the recessed edges of the intact frontal bone. Additionally, the height of both supraorbital rims was reduced to improve the slope of the patient’s forehead. Once a satisfactory contour had been

Discussion PD is a rare condition, and the true incidence remains unknown.1,3 As illustrated by the present case, most patients with PD present electively to surgeons with what they believe to be an isolated cosmetic concern. Thus, it is likely that the true incidence is greater than that represented in published studies. As a result of advancements in imaging technology and an increasing interest in cosmetic surgery, the number of cases reported annually has shown a steady trend upward.2 Although rare, PD is an important diagnosis for surgeons to recognize, because patients can also have a variety of other medical conditions known to be associated with PD.2 These conditions can be serious in nature and need to be ruled out before any intervention is undertaken. The present case highlights the importance of obtaining a CT scan to evaluate for intracranial pathologic features and of performing nasal endoscopy and intraoperative biopsies to evaluate for other pathologic entities. Because these patients might not present

FIGURE 3. BrainLAB sensors set up on the patient after the induction of anesthesia. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

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SURGICAL NAVIGATION TO TREAT PNEUMOSINUS DILATANS

FIGURE 4. The entire deformed segment of frontal bone was removed en bloc to expose the abnormal, hyperaerated frontal sinus cavities. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

FIGURE 5. Osteotomized bone segments, burred down and set in place with titanium plates to re-create a natural contour to the frontal bone. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

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FIGURE 6. Photographs at 6 months postoperatively of the patient in the A, frontal and B, lateral views. Note the improved contour of the brow and the patient’s new hairstyle, who no longer attempts hide his brow. Ricci, Desai, and Vendemia. Surgical Navigation to Treat Pneumosinus Dilatans. J Oral Maxillofac Surg 2015.

to any other physician for treatment, it is imperative the surgeon establish the correct diagnosis of PD and evaluate these patients carefully before initiating any surgical intervention. The present case is also unique in that it illustrates the use of a real-time intraoperative navigation system to correct the patient’s hyperaerated frontal sinuses. For patients with isolated frontal sinus hyperaeration, the treatment involves removal of the anterior table of the frontal sinus and reshaping of the bone.6 It is important when performing this procedure for a benign condition that the posterior table and dura are not violated, given the risk of intracranial infection.6 Historically, the rates of infection have ranged up to 15% in patients who have undergone cranial reconstruction, although this might be lower with newer generation antibiotics.1,7 In the present case, the use of the BrainLAB system enabled the surgeons to determine the extent of the underlying cavity. The device was fairly easy to use and gave the surgical team real-time information during the operation to prevent violation of the posterior table. Before the development of technology such as BrainLAB, only an estimation of the boundaries of the hyperaerated sinus could be made, and osteotomies would be biased toward the midline out of necessity. This likely reduced the quality and volume of bone needed for eventual reconstruction of the anterior

surface. The use of a surgical navigation system for correction of the cosmetic deformity caused by PD of the frontal sinus has proved to allow for a more efficient and safer operation by accurate osteotomy placement with no wasted bone for reconstruction and no accidental intracranial involvement. Acknowledgments The authors would like to thank Anthony LaBruna, MD, for his assistance with this case and for providing the photographs.

References 1. Taub PJ, Narayan P: Surgical navigation technology for treatment of pneumosinus dilatans. Cleft Palate Craniofac J 44:562, 2007 2. Desai NS, Saboo SS, Khandelwal A, Ricci JA: Pneumosinus dilatans: Is it more than an aesthetic concern? J Craniofac Surg 25:418, 2014 3. Appelt EA, Wilhelmi BJ, Warder DE, Blackwell SJ: A rare case of pneumosinus dilatans of the frontal sinus and review of the literature. Ann Plast Surg 43:653, 1999 4. Walker JL, Jones NS: Pneumosinus dilatans of the frontal sinuses: Two cases and a discussion of its aetiology. J Laryngol Otol 116: 382, 2002 5. Suryanarayanan R, Abbott G: Pneumosinus dilatans: Demonstrated by sinus expansion on serial sinus X-rays with discussion of possible aetiology. J Laryngol Otol 121:96, 2007 6. Galie M, Consorti G, Clauser LC, Kawamoto HK: Craniofacial surgical strategies for the correction of pneumosinus dilatans frontalis. J Craniomaxillofac Surg 41:28, 2013 7. Carey ME, Young HF, Rish BL, Mathis JL: Follow-up study of 103 American soldiers who sustained a brain wound in Vietnam. J Neurosurg 41:542, 1974

Correction of a contour deformity associated with frontal pneumosinus dilatans using surgical navigation technology.

Pneumosinus dilatans (PD) is a pathologic hyperaeration of the paranasal sinuses of unknown etiology. Although benign itself, PD has been associated w...
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