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communicate well with patients. Of course, future prospective clinical studies are necessary and should be undertaken to further validate the accuracy of this protocol for guiding the movement of jaw segment during the actual surgery process by comparing the surgical results with virtual surgery planning.
REFERENCES 1. Cevidanes LH, Tucker S, Styner M, et al. Three-dimensional surgical simulation. Am J Orthod Dentofacial Orthop 2010;138:361Y371 2. Xia JJ, Gateno J, Teichgraeber JF. Three-dimensional computer-aided surgical simulation for maxillofacial surgery. Atlas Oral Maxillofac Surg Clin North Am 2005;13:25Y39 3. Uechi J, Okayama M, Shibata T, et al. A novel method for the 3-dimensional simulation of orthognathic surgery by using a multimodal image-fusion technique. Am J Orthod Dentofacial Orthop 2006;130:786Y798 4. Nadjmi N, Mollemans W, Daelemans A, et al. Virtual occlusion in planning orthognathic surgical procedures. Int J Oral Maxillofac Surg 2010;39:457Y462 5. Metzger MC, Hohlweg-Majert B, Schwarz U, et al. Manufacturing splints for orthognathic surgery using a three-dimensional printer. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:e1Ye7 6. Swennen GR, Barth EL, Eulzer C, et al. The use of a new 3D splint and double CT scan procedure to obtain an accurate anatomic virtual augmented model of the skull. Int J Oral Maxillofac Surg 2007;36:146Y152 7. Aboul-Hosn Centenero S, Hernandez-Alfaro F. 3D planning in orthognathic surgery: CAD/CAM surgical splints and prediction of the soft and hard tissues resultsVour experience in 16 cases. J Craniomaxillofac Surg 2012;40:162Y168 8. Swennen GR, Mommaerts MY, Abeloos J, et al. A cone-beam CT based technique to augment the 3D virtual skull model with a detailed dental surface. Int J Oral Maxillofac Surg 2009;38:48Y57 9. Xia JJ, Gateno J, Teichgraeber JF. New clinical protocol to evaluate craniomaxillofacial deformity and plan surgical correction. J Oral Maxillofac Surg 2009;67:2093Y2106
Perioperative Difficulties and Early Postoperative Complications of Transoral Approach in Mouth Base Surgery Ercan Akbay, MD, Cengiz Cevik, MD, Cengiz Arli, MD Objective: The aim of this study was to discuss perioperative difficulties and temporary or permanent complications that can be seen at early postoperative period in cases undergoing transoral surgical approach for disorders of mouth base. From the Department of Otorhinolaryngology Head & Neck Surgery, Mustafa Kemal University Medical Faculty, Hatay, Turkey. Received May 16, 2013. Accepted for publication September 17, 2013. Address correspondence and reprint requests to Ercan Akbay, MD, Mustafa ¨ niversitesi Tip Faku¨ltesi KBB Anabilim Dali, Serinyol, Hatay, Kemal U Turkey; E-mail: [email protected] The authors report no conflicts of interest The author obtained written informed consent from the patients for submission of this article for publication. Authors’ contributions: E.A.: idea, surgical approach, writing, and correspondence. C.C.: surgical approach. C.A.: writing. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000413
Brief Clinical Studies
Study Design: The study included 19 patients undergoing transoral surgical approach between September 2011 and January 2013 who were retrospectively evaluated. Exclusion criteria were patients with malign submandibular gland tumor or other suspected tumors at mouth base, those with a benign solid mass larger than 10 10 cm, and those who did not accept the transoral approach. Results: Mean age was 28.47 T 17.89 years (range, 8Y76 years) in 19 patients included (9 male and 10 female subjects). Of these, submandibular gland excision was performed in 9 cases, whereas thyroglossal duct cyst excision was performed in 3 (2 transfrenulum approach and 1 lateral lingual approach), lymphangioma excision in 1, diagnostic lymph node excision at the posterior of submandibular gland in 3 cases, and dermoid cyst excision (2 with midline and 1 with left submandibular localization) in 3 cases. Perioperative difficulties included dissection problems due to adhesion, partial adhesion between Wharton canal and lingual nerve, fragmented dissection of the gland, capsule rupture of submandibular adenoma, and facial artery rupture. While visualization of surgical field and retraction of mouth base muscles are an important issue in midline approaches, resection of hyoid bone corpus is challenging in lateral approaches. Early postoperative complications included edema at mouth base, lingual ecchymosis, and postoperative temporary abnormal tongue sensation. Conclusions: Transoral mouth base surgery is a safe approach in selected patients. Permanent injuries of neural structures are rarely encountered, which is considered as a concern by head and neck surgeons. Temporary complications are at a level that can be tolerated by patients. Key Words: Mouth base surgery, transoral approach, submandibular gland, lymphangioma, thyroglossal cyst, dermoid cyst
T
he patients undergoing paralingual, paraglandular, submandibular, submental, and parahyoid surgeries were classified as mouth base surgery in the current study. These surgeries included those involving the submandibular gland and duct, thyroglossal duct cysts, submandibular lymphangioma, dermoid cysts, and diagnostic lymph node biopsies. The common feature is that all surgical interventions are the procedures performed in benign diseases or excision biopsies, all of which were performed through the transoral route. Submandibular gland excisions are generally performed in the indications including neoplasm, chronic sialadenitis, and sialalithiasis.1,2 The traditional treatment for submandibular diseases is open-field sialoadenectomy, which is based on the transcervical approach.3,4 Traditionally, transcervical approach has been used in the submandibular lymphangiomas localized under the mylohyoid muscle as well. It is also used in thyroglossal duct cysts causing swelling at midline in the neck and recurrent infections. In recent years, transoral approach has been increasingly used in mouth base surgeries including mainly submandibular gland excision as well as thyroglossal duct cysts and dermoid cysts.5Y8 In particular, these approaches become more important in young women and children because of undesirable external skin incision and scar tissue. It was shown that 29% of salivary calculi are intraglandular, whereas 71% are intraductal in the literature.9 In case of a calculus distal to Wharton canal, transcervical approach may fail. Thus, exploration and resection of duct in combination with gland may be performed by transoral route. Transoral submandibular gland excisions, first described by Downton and Qvist in the 1960s, gained popularity by development of endoscopy and other surgical techniques.1 Although it has been initially used only in chronic sialadenitis, it has been used in benign submandibular gland tumors later.10
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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FIGURE 1. In the excisional biopsy of paraglandular lymph nodes localized at the submandibular region, Wharton canal, lingual nerve, hypoglossal nerve, gland, and digastrics muscles were also identified as in submandibular gland excision, and surgical approach was performed by sparing these structures (a: lymphadenopathy, b: lingual nerve, c: Wharton duct).
So et al6 and Kim et al7 described transoral-transfrenulum approach in the surgery of thyroglossal duct cysts. In our department, we performed thyroglossal duct excision by transoral lateral lingual approach in addition to transfrenulum approach. Sublingual dermoid cysts localized on the mylohyoid muscle have been long treated via the transoral approach. In parallel to sociocultural development, aesthetic concerns and expectation are increasing worldwide. We believe that the transoral approach will be widely used over time, which is compelling in terms of cosmetic outcomes. However, it is associated with some difficulties in practice that make surgeons feel uncomfortable and postoperative complications. In the current study, we discussed patients who underwent transoral surgery, surgical difficulties in these patients, recommendations, and temporary or permanent complications that can be seen at the early postoperative period.
PATIENTS AND METHODS The patients, who underwent transoral surgical approaches with initial diagnoses thyroglossal duct cyst, dermoid cyst, lymphangioma, lymphadenopathy, benign submandibular gland tumor, chronic sialadenitis, and sialalithiasis between September 2011 and January 2013, were retrospectively evaluated. The patients undergoing surgeries because of lymphadenopathy were selected among those scheduled for diagnostic excision. The patients with malign submandibular gland tumor or other suspected tumors at mouth base, those with a benign solid mass larger than 10 10 cm, and those who did not accept the transoral approach were excluded. All patients underwent neck sonography, whereas computed tomography scanning was performed in the patients with suspected malignity. All patients were informed that the procedure could be converted to a conventional wide-open transcervical approach in case of problematic conditions that cannot be accomplished via the transoral approach. All patients underwent surgery under general anesthesia by using nasotracheal intubation. The patients were placed in the supine position, with the neck semiextended and the head laterally rotated to the contralateral side. The surgeon stood at one side of the patients, and the assistant and scrub nurse stood at the left and right side of the patient. Endoscope (30-degree 4-mm rigid endoscope; Karl Storz Tuttlingen, Germany) was used in all patients, as it provides improved lightening, magnification of surgical field, and video capturing. The Digman mouth gag (Fixity Surgical Industries, Punjab, Pakistan) was placed into the oral cavity in an inverted fashion, and 2 tongue retractors were used to expose the mouth base. One percent lidocaine with a 1:100,000 dilution of epinephrine was injected to the mouth base mucosa. For the submandibular gland excision, a mucosal incision was made extending from the anterior of the Wharton canal to the
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retromolar trigone at approximately 1 cm medial to gingival, passing parallel to canal. At this point, sharp dissections were avoided to prevent injury to neurovascular structures, and blunt dissections were used. First, Wharton canal was identified and traced until the point where it crosses the lingual nerve. The lingual nerve was meticulously dissected by avoiding the deterioration of surgical exposure. Submandibular ganglion could be seen in some cases at the point where it branches from the lingual nerve; however, it might be disrupted in some cases as it is very fragile. The Wharton canal was divided by sparing the sublingual gland duct, and it was passed under the lingual nerve, which was then retracted posteriorly. Thus, a submandibular gland exposure was achieved that involved no nerve between the gland and canal. The submandibular gland is then progressively separated from the inferior border of the mylohyoid, the genioglossus, and the hyoglossus muscles by blunt dissections. At this point, the hypoglossal nerve could be identified in some cases, but it might be seen with a fascia-like structure overlying the nerve in a closed manner, and the hypoglossal nerve could be seen after dissection of this fascia-like structure in others. However, we did not observe the hypoglossal nerve as being in the gland or adherent to gland in any case including those with chronic sialadenitis. The facial artery and vein were identified at the posterolateral localization, which were clamped and secured, if indicated. However, the small branches of the facial artery and vein may rupture during blunt dissection, and intensive bleeding may occur for a while. These bleedings may spontaneously stop when a direct compression is applied by using gauze. After controlling the hemorrhage and neurovascular structures, surgical cavity was flushed by normal saline and closed via interrupted 4-0 rapid Vicryl sutures by leaving a small opening at the posterior for drainage. We also used the transoral lateral lingual approaches in the excision of dermoid cysts with a lateral localization, excision of submandibular lymphangioma, biopsies for lymphadenopathy (Fig. 1), and surgery of thyroglossal duct cysts localized at midline. However, as in submandibular gland excision, the Wharton canal, lingual nerve, hypoglossal nerve, gland, and digastric muscles were identified, and the target mass or cyst was excised by sparing these structures. In the excision of large masses localized under the mylohyoid muscle, the muscle is incised by a bipolar cautery device to facilitate dissection. In the midline surgeries, transfrenulum incision was made to excise dermoid cysts and thyroglossal duct cysts in particular. In case of dermoid cyst, the capsule of the cyst may be encountered after mucosal incision. The retraction of geniohyoid muscles may be needed in the dermoid cyst with more profound localization (Fig. 2). In case of thyroglossal duct cysts, dissection was advanced between genioglossus muscles after mucosal incision, and hyoid bone corpus was accessed by tracing the geniohyoid muscles. In the midline approach to a mass lesion, blunt dissection was preferred throughout the surgery after mucosal incision. Bone scissors was used at the point where the hyoid bone corpus was excised, whereas monopolar cautery device was used in the excision of the muscles adherent to hyoid bone.
FIGURE 2. In the dermoid cysts with midline and profound localization, it is difficult to ensure sufficient visualization despite retraction of genioglossus and geniohyoid muscles.
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RESULTS The mean age was 28.47 T 17.89 years (range, 8Y76 years) in 19 patients (9 men and 10 women) who were included (Table 1). Of these, submandibular gland excision was performed in 9 cases, whereas thyroglossal duct cyst excision was performed in 3 (2 transfrenulum approach and 1 lateral lingual approach), lymphangioma excision from submandibular region in 1, diagnostic lymph node excision at the posterior of submandibular gland in 3, and dermoid cyst excision (2 with midline and 1 with left submandibular localization) in 3 cases. Of the dermoid cysts, 2 were localized at submental region, whereas the other
Brief Clinical Studies
was localized at the left submandibular region. Mean surgery time was measured as 77.63 T 18.88 minutes (range, 45Y110 minutes). The shortest surgery time was recorded as 45 minutes in a midline dermoid cyst, whereas the longest surgery time was recorded as 110 minutes in a pleomorphic adenoma of the submandibular gland. The primary difficulty during surgery was dissection due to adhesion in the presence of limited exposure, which was particularly observed in 3 cases with chronic sialadenitis. Adhesions were observed at the anterior and anterolateral aspects of the gland in all submandibular gland excisions associated to difficulty in dissection due to adhesion. Of these, gland was excised in a fragmented manner
TABLE 1. Distribution of the Patients Who Underwent Mouth Base Surgeries by Transoral Approach in Terms of Clinical Diagnosis, Surgical Procedure, Perioperative Difficulties, Early Postoperative Complications, Pathological Results, and Surgery Time
No. Age,
Sex
Initial Diagnoses
1
9
Male
2
38
Female Right submandibular sialadenitis
3
76
Female Right submandibular sialadenitis
4
56
Female Left submandibular sialadenitis
5
39
Male
Left submandibular sialadenitis
6
12
Male
Left submandibular sialadenitis
7
30
Female Left submandibular sialadenitis
8
36
Female Right submandibular sialadenitis
9
35
Female Left submandibular gland adenoma 2.5 3 cm
10
19
11
31
12
32
13
45
14
10
15
22
16 17
8 16
Male Thyroglossal duct cyst Female Thyroglossal duct cyst
18 19
13 14
Female Thyroglossal duct cyst Male Left submandibular lymphangioma 6 9 cm
Left submandibular sialadenitis
Male
Surgical Approach Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland and mass excision Transoral transfrenulum
Perioperative Difficulty and Complication
Early Postoperative Complications
None
None
Chronic sialadenitis
80
None
Chronic sialadenitis
90
Chronic sialadenitis with acute episodes
70
Chronic sialadenitis + sialalithiasis
90
Fragmented dissection of the gland due to adhesion Facial artery rupture
Difficult dissection of lingual nerve and Wharton duct due to adhesion Difficult dissection of gland due to adhesion
Edema at submandibular region and lingual ecchymosis Temporary abnormal tongue sensation
Left submandibular conglomerate lymphadenopathy Female Right submandibular lymphadenopathy
None
None
Surgery Time, min
100
None
None
Chronic sialadenitis
65
Fragmented dissection of the gland + facial artery rupture
None
Chronic sialadenitis
80
None
None
Chronic sialadenitis + sialalithiasis
65
None
Submandibular gland pleomorphic adenoma
None
Dermoid cyst
45
None
Dermoid cyst
50
None
Dermoid cyst
60
None
85
90 100 100 80
Rupture of adenoma capsule due to excessive size of mass
Submental dermoid Challenging dissection cyst 8 10 cm without causing rupture Female Submental dermoid Transoral transfrenulum None cyst 8 9 cm Male Left submandibular Transoral Elongation and dermoid cyst 5 8 cm endoscopy-assisted deformation of the duct cyst excision Female Left submandibular Transoral retroglandular Difficult exposure and dissection lymphadenopathy approach Male
Histopathology
Transoral retroglandular lymph node excision
None
None
Transoral retroglandular lymph node excision
None
None
Transoral transfrenulum Transoral lateral lingual approach Transoral transfrenulum Transoral lymphangioma and submandibular gland excision
Difficult exposure and dissection Difficult excision of hyoid bone Difficult exposure and dissection Difficult dissection inferior to mylohyoid muscle and rupture of the capsule
Submental edema None
Low-grade non-Hodgkin lymphoma Granulomatous lymphadenitis with necrosis Chronic granulomatous inflammation with caseification necrosis Thyroglossal duct cyst Thyroglossal duct cyst
Submental edema None
Thyroglossal duct cyst Lymphangioma
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60
55
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FIGURE 3. The gland can be removed in a fragmented manner in case of difficult dissection due to adhesion during submandibular gland excision.
in 2 cases (Fig. 3), whereas facial artery rupture occurred in another 2 cases, which were then ligated (Fig. 4). Capsule of the mass was ruptured in a case with submandibular adenoma, whereas dissection became more difficult because of rupture risk related to the extent of the cyst in dermoid cysts. A postoperative temporary abnormal tongue sensation was detected due to partial adhesion observed between Wharton canal and lingual nerve in a patient who underwent surgery for sialalithiasis (Fig. 5). Postoperative mouth base edema was detected in 2 cases with thyroglossal duct cyst, whereas postoperative mouth base edema and lingual ecchymosis were observed in a submandibular gland excision (Fig. 6). The cystic mass was localized inferior to the mylohyoid muscle in a case with lymphangioma, in which excision was performed via the transoral approach (Fig. 7). The exposure was improved by partial dissection of the muscle by using a bipolar electrocautery device (Fig. 8). The cyst rupture occurred despite this process. However, when ruptured cyst was evaluated, it was seen that cyst integrity was not disrupted, and en bloc cyst excision was possible. While visualization of surgical field and retraction of mouth base muscles were important problems in the thyroglossal duct cysts excised via midline approach (Fig. 9), resection of hyoid bone corpus was challenging in the lateral lingual approach (Fig. 10). A deformation was observed in the submandibular canal that resulted from the expansion of cystic mass and abnormal anatomical localization during the excision of dermoid cyst with lateral localization (Fig. 11).
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FIGURE 5. A partial adhesion is observed between Wharton canal and lingual nerve in a case with previous duct inflammation due to sialalithiasis (a: deformed Wharton duct containing a calculus, b: lingual nerve).
Partial synechia between Wharton canal and lingual nerve is one of the perioperative difficulties that can be observed in cases with sialadenitis, particularly in those with sialalithiasis. The nerve can be readily removed from duct by a meticulous dissection. Although the nerve does not rupture during this dissection, temporary abnormal tongue sensation or hypoesthesia may be observed. However, similar lingual paresthesia may be encountered in transcervical approaches in case of chronic sialalithiasis, if severe tissue adhesions are present.9,11 Weber et al2 found the rate of postoperative temporary abnormal tongue sensation as 43% in transoral surgical approaches, which were performed by leaving sufficient tissue in gingival mucosa to close the surgical field without excessive skeletonization of lingual nerve. In our case series where patients underwent similar surgical procedures, abnormal tongue sensation was observed in only 1 patient, which resolved at week 4 after surgery. In this case, a
mild adhesion was observed at the point where lingual nerve crosses the submandibular duct and dissection was hardly achieved. In cases with chronic sialadenitis, submandibular gland dissection can be rather difficult because of adhesion especially in the presence of previous abscess history. The adhesions were frequently observed in the anterolateral aspect of the gland, but no adhesion was observed in the base of gland or adjacent to the hypoglossal nerve. In these cases, the gland can be removed in a fragmented manner. In our study, the gland was excised in a fragmented manner in 2 cases. However, no gland tissue was left in the submandibular space. Again, difficulties in dissection did not cause paresthesia in the marginal mandibular branch of the facial nerve. Facial artery rupture is another perioperative complication that is rarely presented as a risk during blunt dissections. Facial artery rupture occurred in 2 cases in our study; however, bleeding was readily controlled by clamping the artery. Surgical clips can be used for this purpose; however, ligation of the facial artery in the transoral approach is not difficult for the surgeons performing classic tonsillectomy and preferring to ligate the tonsillary artery. Another concern is contamination or infection of the surgical field by oral flora. We use perioperative irrigation with chlorhexidine gluconate and administer antibiotic covering oral flora to reduce risk. However, there is a risk like this in cases undergoing tonsillectomy. Postoperative surgical infections are rarely observed in patients who underwent tonsillectomy; similarly, we observed no infection in patients who underwent surgeries via the transoral approach. There is a risk for perioperative paresthesia or paralysis of the marginal mandibular branch of the facial nerve, which is lower than that of transcervical approach, because it is localized in the subplatysmal plane, which protects it during transoral approach.1 Minimal invasive video-assisted sialoadenectomies are performed to avoid complications specific to transoral approach and scar tissue related to transcervical incision.3,12,13 However, temporary paresthesia of the marginal mandibular branch of facial nerve can be seen in these procedures as well.3 The transoral procedure performed in benign submandibular tumors such as adenoma is one of the factors that make perioperative surgical dissection difficult. In such cases, manipulation of a large mass in a limited area and en bloc dissection could be difficult. Moreover, integrity of the mass capsule can be disrupted. However, it is possible not to experience a recurrence if surgical field is sufficiently
FIGURE 4. Ruptured facial artery can be easily identified, which is then clamped and ligated (a: ruptured facial artery, b: hypoglossal nerve).
FIGURE 6. Lingual ecchymosis and edema at early postoperative period were seen in an elder patient with hypertension.
DISCUSSION
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FIGURE 7. Preoperative (A) and postoperative appearance (B) of a patient with lymphangioma excised by transoral route without an external incision.
eliminated without leaving residual tissue. Because it is known that the integrity of the capsule is occasionally disrupted during parotid gland surgery, no recurrence is observed unless any residual tissue is left, and the surgical field is sufficiently eliminated by irrigation. There is a rupture risk during dissection of cystic masses. The risk further increases in the transoral approach because of limited visualization and dissection space. We perform blunt dissection by placing angled clamps or angled part of a small retractor to the base of cyst especially in the dissection of the base. We did not observe any rupture even in the dermoid cysts up to 8 10 cm by using the previously mentioned process. The Sistrunk operation is the traditional surgical treatment in thyroglossal cysts, which is achieved by transcervical midline incision. Although the traditional approach is the only method in thyroglossal fistulas and in case of fistula formation between cyst and skin, the transoral approach can be preferred in case of undesirable skin incision or cysts without fistula formation. We observed diffuse edema that does not cause respiratory distress in patients who underwent surgery via the transoral approach. Alternatively, we used the lateral lingual approach in 1 case. We accessed the hyoid bone and cyst by tracing the digastric and hyoglossus muscles after exposing and sparing the submandibular gland and hypoglossal nerve. We observed less edema at the mouth base in this case. Another postoperative complication is edema at the mouth base and tongue, causing problems in mastication and swallowing after surgery. Increased edema due to trauma resulted from manipulation of muscles by retractors, and surgery may lead to scar tissue and contracture resulting in limitation of tongue movement in the long term. We make effort to not traumatize the muscles during surgery to reduce postoperative edema. We apply compression bandage to the submandibular or sublingual region at the end of surgery. In addition, we apply external cold by using cold packages and recommend drinking cold beverages. However, we did not observe edema at the tongue and mouth base sufficient to cause respiratory distress in any patient. The lateral approach is used in the submandibular gland excisions, and muscle dissection is limited in this approach other than retraction of mylohyoid muscle to anterolateral in a limited area. However, in the midline transfrenulum approach used in thyroglossal duct cysts, genioglossus and geniohyoid muscles are retracted by using retractors in the profound plane in particular. Thus, edema encountered is more massive in midline than in lateral approaches. Although midline approaches have the disadvantage of traumatizing muscles, it has the advantages such as lack of neurovascular structures. Another concern is laceration of the Wharton canal and caruncula in the surgery of thyroglossal duct and dermoid cysts via transoral midline incisions. This problem is not frequently observed
FIGURE 8. Mylohyoid muscle was partially dissected by bipolar electrocautery (A) and cystic mass was exposed (B).
Brief Clinical Studies
FIGURE 9. For transoral midline excisions of the thyroglossal cyst, the genioglossus muscles are retracted to lateral first, and the hyoid bone was accessed by tracing geniohyoid muscles, which has more profound localization (A) (a: genioglossus muscle, b: geniohyoid muscle, c: thyroglossal duct cyst). Preoperative (B) and postoperative appearance (C) of thyroglossal duct cyst without fistula formation between the cyst and skin.
in cases in which localization is achieved by the observation of saliva output through the caruncula after Wharton massage. Alternatively, this problem can be solved by initiating the first incision at a point over the frenulum close to the lingual end and advancing by blunt dissections close to caruncula. Dermoid cysts are commonly observed teratomatous congenital mass with benign features. Of all the dermoid cysts, 1% to 7% is detected in the head-neck region; of these, 23% are observed at the mouth base.14 The cyst is clinically classified into 3 types based on its relationship to muscles of the mouth base and the geniohyoid and mylohyoid muscles.15 The more common type is localized above the geniohyoid and mylohyoid muscles, which are clinically visible in the mouth base as they displace the tongue upward. If it is localized between the geniohyoid and the mylohyoid muscle or inferior to the mylohyoid muscle, it would appear as double chin. The third type is localized at an area where the cyst is displaced laterally into the submandibular area.16 In our clinic, we used excision via the transoral approach in all types. Total excision without disrupting capsule integrity is essential to avoid recurrences. In transoral approach, the problem is to achieve the excision in a limited area. We used blunt dissection throughout the surgery except the initial mucosal incision. Serious complications such as respiratory distress are seen in the untreated lymphangiomas localized at the neck and submandibular region.17 Sclerosing agents or surgical therapies can be used in the treatment of the lymphangiomas localized at this region.18 Recurrence has been reported in cases treated by injections of sclerosing agents.17 However, surgical therapies have undesirable outcomes such as incisions causing scar tissue. We excised a submandibular lymphangioma localized inferior to the mylohyoid muscles via the transoral approach. By this approach, cystic mass was totally resected without creating an incision scar at the skin. The successful removal of the lymphangioma despite difficult dissection due to limited visualization was a satisfactory outcome for patients. In our study, perioperative problems during diagnostic lymph node excision at the submandibular region included those related to identification of neurovascular structures and Wharton canal, which were observed in a similar appearance as in submandibular gland excision; in addition, it also included limited visualization and difficult dissection resulting from more posterior localization in retroglandular lymph node dissections. Endoscopic dissection is
FIGURE 10. Although more limited muscle retraction is performed in the lateral lingual approach to thyroglossal duct cyst, hyoid bone corpus can be barely resected, and it is warranted to expose neurovascular structures (a: hyoglossus muscle, b: lingual nerve, c: Wharton duct).
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FIGURE 11. Deformation in submandibular canal and abnormal anatomical localization are seen in a dermoid cyst with lateral localization (A) (a: lingual nerve, b: duct of sublingual gland, c: common duct, d: Wharton duct). Neural and ductal structures are seen to be spared after the excision of the mass (B) (a: lingual nerve, b: Wharton duct, c: duct of sublingual gland).
helpful at this point. Early postoperative complications were similar to those seen in the submandibular gland excision. In conclusion, the transoral mouth base surgery is a safe surgical approach in selected patients. Permanent injuries of neural structures are rarely encountered, which is considered as a concern by head and neck surgeons. The hypoglossal nerve is located at the base of the submandibular gland; it is generally separated from the gland by a fascia-like soft tissue. Also, one cannot readily injure the hypoglossal nerve because of its thickness and durability, unless sharp dissection is used. Although the lingual nerve occasionally presents with adhesion to duct in cases with sialadenitis, no permanent injury is seen by meticulous dissection. Temporary complications and especially abnormal tongue sensations are well tolerated by the patients.
REFERENCES 1. Kauffman RM, Netterville JL, Burkey BB. Transoral excision of the submandibular gland: techniques and results of nine cases. Laryngoscope 2009;119:502Y507 2. Weber SM, Wax MK, Kim JH. Transoral excision of the submandibular gland. Otolaryngol Head Neck Surg 2007;137:343Y345 3. Ruscito P, Pichi B, Marchesi P, et al. Minimally invasive video-assisted submandibular sialoadenectomy: a preliminary report. J Craniofac Surg 2007;18:1142Y1147 4. Chen WL, Yang ZH, Wang YJ, et al. Removal of the submandibular gland using a combined retroauricular and transoral approach. J Oral Maxillofac Surg 2009;67:522Y527 5. Chukwuneke FN, Akaji C, Onyeka TC, et al. Surgical excision of intra-oral dermoid cyst under local anaesthesia: a review of nine cases. J Maxillofac Oral Surg 2010;9:19Y21 6. So YK, Jeong JI, Youm HY, et al. Endoscope-assisted intra-oral resection of the external thyroglossal duct cyst. Am J Otolaryngol 2011;32:71Y74 7. Kim JP, Park JJ, Lee EJ, et al. Intraoral removal of a thyroglossal duct cyst using a frenotomy incision. Thyroid 2011;21:1381Y1384 8. Beahm DD, Peleaz L, Nuss DW, et al. Surgical approaches to the submandibular gland: a review of literature. Int J Surg 2009;7:503Y509 9. Ellies M, Laskawi R, Arglebe C, et al. Surgical management of nonneoplastic diseases of the submandibular gland. A follow-up study. Int J Oral Maxillofac Surg 1996;25:285Y289 10. Hong KH, Yang YS. Intraoral approach for the treatment of submandibular salivary gland mixed tumors. Oral Oncol 2008;44:491Y495 11. Baek CH, Jeong HS. Endoscope-assisted submandibular sialadenectomy: a new minimally invasive approach to the submandibular gland. Am J Otolaryngol 2006;27:306Y309 12. Komatsuzaki Y, Ochi K, Sugiura N, et al. Video-assisted submandibular sialadenectomy using an ultrasonic scalpel. Auris Nasus Larynx 2003;30 Suppl:S75YS78 13. Hamza Y, Khalil R. Video-assisted submandibular resection: two-step technique. Surg Endosc 2009;23:2785Y2789 14. Go¨ru¨r K, Talas DU, Ozcan C. An unusual presentation of neck dermoid cyst. Eur Arch Otorhinolaryngol 2005;262:353Y355 15. Meyer I. Dermoid cysts (dermoids) of the floor of the mouth. Oral Surg Oral Med Oral Pathol 1955;8:1149Y1164
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16. Zachariades N, Skoura-Kafoussia C. A life-threatening epidermoid cyst of the floor of the mouth: report of a case. J Oral Maxillofac Surg 1990;48:400Y403 17. Grasso DL, Pelizzo G, Zocconi E, et al. Lymphangiomas of the head and neck in children. Acta Otorhinolaryngol Ital 2008;28:17Y20 18. Gigue`re CM, Bauman NM, Sato Y, et al. Treatment of lymphangiomas with OK-432 (Picibanil) sclerotherapy: a prospective multi-institutional trial. Arch Otolaryngol Head Neck Surg 2002;128:1137Y1144
Aneurysmal Bone Cyst of the Zygomatic Bone Jun Yong Lee, MD, Young-Il Ko, MD, Ho Kwon, MD, Sung-No Jung, MD Abstract: Aneurysmal bone cyst is a rare, non-neoplastic lesion that mostly involves the long bone and the spine, and is characterized by its expansile, vascular, and multi-cystic features. Reports of facial bone lesions are rare, and when it occurs, is usually located in the mandible. Herein, this report is aimed to describe a very rare case of an aneurysmal bone cyst in the zygoma with a brief review of the literature. Key Words: Aneurysmal bone cyst, zygomatic bone, neoplasm, bone tumor
A
neurysmal bone cyst is a septated pseudocyst that is devoid of epithelial lining and comprises a blood-filled cavity.1 It is commonly found in the long bone and the spine, and its incidence in the facial bone is rare, with most of these cases occurring in the mandible; 3 cases involving the zygoma have been reported to date.2 A 45-year-old female patient visited our outpatient department with a chief complaint of a left malar nodule that had developed about 15 months before presentation. There was no relevant history such as preceding facial trauma, and physical examination showed a nontender, hard, palpable mass on her left malar area. Computed tomography (CT) images revealed an intrabony, expansile lesion, with marked thinning of the surrounding zygoma (Fig. 1). No internal calcification within the mass was evident. Under general anesthesia, the authors completely excised the lesion through a subciliary incision; the resultant bone defect was repaired by filling in with bone putty (Allomatrix; Wright Medical Technology, Inc, Arlington, TN) (Fig. 2). Subsequent histopathologic examination showed a sinusoidal space filled with erythrocytes, fibroblasts, and histiocytes in a fibrous matrix (Fig. 3). No newly developed lesion was seen on a follow-up CT imaging performed 12 months postoperatively (Fig. 4), and the patient is currently on constant follow-up for 12 months without any signs of recurrence. From the Department of Plastic and Reconstructive Surgery, Uijeongbu St. Mary’s Hospital College of Medicine Catholic University of Korea, Seoul, Republic of Korea. Received August 08, 2013. Accepted for publication September 16, 2013. Address correspondence and reprint request to Sung-No Jung, M.D, Department of Plastic and Reconstructive Surgery, Uijeongbu St Mary’s Hospital, College of Medicine, Catholic University of Korea, 271 Cheonbo-ro, Uijeongbu-si, Gyeonggi-do, 480-717, Seoul, Republic of Korea; E-mail:[email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000463
* 2014 Mutaz B. Habal, MD
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communicate well with patients. Of course, future prospective clinical studies are necessary and should be undertaken to further validate the accuracy of this protocol for guiding the movement of jaw segment during the actual surgery process by comparing the surgical results with virtual surgery planning.
REFERENCES 1. Cevidanes LH, Tucker S, Styner M, et al. Three-dimensional surgical simulation. Am J Orthod Dentofacial Orthop 2010;138:361Y371 2. Xia JJ, Gateno J, Teichgraeber JF. Three-dimensional computer-aided surgical simulation for maxillofacial surgery. Atlas Oral Maxillofac Surg Clin North Am 2005;13:25Y39 3. Uechi J, Okayama M, Shibata T, et al. A novel method for the 3-dimensional simulation of orthognathic surgery by using a multimodal image-fusion technique. Am J Orthod Dentofacial Orthop 2006;130:786Y798 4. Nadjmi N, Mollemans W, Daelemans A, et al. Virtual occlusion in planning orthognathic surgical procedures. Int J Oral Maxillofac Surg 2010;39:457Y462 5. Metzger MC, Hohlweg-Majert B, Schwarz U, et al. Manufacturing splints for orthognathic surgery using a three-dimensional printer. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:e1Ye7 6. Swennen GR, Barth EL, Eulzer C, et al. The use of a new 3D splint and double CT scan procedure to obtain an accurate anatomic virtual augmented model of the skull. Int J Oral Maxillofac Surg 2007;36:146Y152 7. Aboul-Hosn Centenero S, Hernandez-Alfaro F. 3D planning in orthognathic surgery: CAD/CAM surgical splints and prediction of the soft and hard tissues resultsVour experience in 16 cases. J Craniomaxillofac Surg 2012;40:162Y168 8. Swennen GR, Mommaerts MY, Abeloos J, et al. A cone-beam CT based technique to augment the 3D virtual skull model with a detailed dental surface. Int J Oral Maxillofac Surg 2009;38:48Y57 9. Xia JJ, Gateno J, Teichgraeber JF. New clinical protocol to evaluate craniomaxillofacial deformity and plan surgical correction. J Oral Maxillofac Surg 2009;67:2093Y2106
Perioperative Difficulties and Early Postoperative Complications of Transoral Approach in Mouth Base Surgery Ercan Akbay, MD, Cengiz Cevik, MD, Cengiz Arli, MD Objective: The aim of this study was to discuss perioperative difficulties and temporary or permanent complications that can be seen at early postoperative period in cases undergoing transoral surgical approach for disorders of mouth base. From the Department of Otorhinolaryngology Head & Neck Surgery, Mustafa Kemal University Medical Faculty, Hatay, Turkey. Received May 16, 2013. Accepted for publication September 17, 2013. Address correspondence and reprint requests to Ercan Akbay, MD, Mustafa ¨ niversitesi Tip Faku¨ltesi KBB Anabilim Dali, Serinyol, Hatay, Kemal U Turkey; E-mail: [email protected] The authors report no conflicts of interest The author obtained written informed consent from the patients for submission of this article for publication. Authors’ contributions: E.A.: idea, surgical approach, writing, and correspondence. C.C.: surgical approach. C.A.: writing. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000413
Brief Clinical Studies
Study Design: The study included 19 patients undergoing transoral surgical approach between September 2011 and January 2013 who were retrospectively evaluated. Exclusion criteria were patients with malign submandibular gland tumor or other suspected tumors at mouth base, those with a benign solid mass larger than 10 10 cm, and those who did not accept the transoral approach. Results: Mean age was 28.47 T 17.89 years (range, 8Y76 years) in 19 patients included (9 male and 10 female subjects). Of these, submandibular gland excision was performed in 9 cases, whereas thyroglossal duct cyst excision was performed in 3 (2 transfrenulum approach and 1 lateral lingual approach), lymphangioma excision in 1, diagnostic lymph node excision at the posterior of submandibular gland in 3 cases, and dermoid cyst excision (2 with midline and 1 with left submandibular localization) in 3 cases. Perioperative difficulties included dissection problems due to adhesion, partial adhesion between Wharton canal and lingual nerve, fragmented dissection of the gland, capsule rupture of submandibular adenoma, and facial artery rupture. While visualization of surgical field and retraction of mouth base muscles are an important issue in midline approaches, resection of hyoid bone corpus is challenging in lateral approaches. Early postoperative complications included edema at mouth base, lingual ecchymosis, and postoperative temporary abnormal tongue sensation. Conclusions: Transoral mouth base surgery is a safe approach in selected patients. Permanent injuries of neural structures are rarely encountered, which is considered as a concern by head and neck surgeons. Temporary complications are at a level that can be tolerated by patients. Key Words: Mouth base surgery, transoral approach, submandibular gland, lymphangioma, thyroglossal cyst, dermoid cyst
T
he patients undergoing paralingual, paraglandular, submandibular, submental, and parahyoid surgeries were classified as mouth base surgery in the current study. These surgeries included those involving the submandibular gland and duct, thyroglossal duct cysts, submandibular lymphangioma, dermoid cysts, and diagnostic lymph node biopsies. The common feature is that all surgical interventions are the procedures performed in benign diseases or excision biopsies, all of which were performed through the transoral route. Submandibular gland excisions are generally performed in the indications including neoplasm, chronic sialadenitis, and sialalithiasis.1,2 The traditional treatment for submandibular diseases is open-field sialoadenectomy, which is based on the transcervical approach.3,4 Traditionally, transcervical approach has been used in the submandibular lymphangiomas localized under the mylohyoid muscle as well. It is also used in thyroglossal duct cysts causing swelling at midline in the neck and recurrent infections. In recent years, transoral approach has been increasingly used in mouth base surgeries including mainly submandibular gland excision as well as thyroglossal duct cysts and dermoid cysts.5Y8 In particular, these approaches become more important in young women and children because of undesirable external skin incision and scar tissue. It was shown that 29% of salivary calculi are intraglandular, whereas 71% are intraductal in the literature.9 In case of a calculus distal to Wharton canal, transcervical approach may fail. Thus, exploration and resection of duct in combination with gland may be performed by transoral route. Transoral submandibular gland excisions, first described by Downton and Qvist in the 1960s, gained popularity by development of endoscopy and other surgical techniques.1 Although it has been initially used only in chronic sialadenitis, it has been used in benign submandibular gland tumors later.10
* 2014 Mutaz B. Habal, MD
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FIGURE 1. In the excisional biopsy of paraglandular lymph nodes localized at the submandibular region, Wharton canal, lingual nerve, hypoglossal nerve, gland, and digastrics muscles were also identified as in submandibular gland excision, and surgical approach was performed by sparing these structures (a: lymphadenopathy, b: lingual nerve, c: Wharton duct).
So et al6 and Kim et al7 described transoral-transfrenulum approach in the surgery of thyroglossal duct cysts. In our department, we performed thyroglossal duct excision by transoral lateral lingual approach in addition to transfrenulum approach. Sublingual dermoid cysts localized on the mylohyoid muscle have been long treated via the transoral approach. In parallel to sociocultural development, aesthetic concerns and expectation are increasing worldwide. We believe that the transoral approach will be widely used over time, which is compelling in terms of cosmetic outcomes. However, it is associated with some difficulties in practice that make surgeons feel uncomfortable and postoperative complications. In the current study, we discussed patients who underwent transoral surgery, surgical difficulties in these patients, recommendations, and temporary or permanent complications that can be seen at the early postoperative period.
PATIENTS AND METHODS The patients, who underwent transoral surgical approaches with initial diagnoses thyroglossal duct cyst, dermoid cyst, lymphangioma, lymphadenopathy, benign submandibular gland tumor, chronic sialadenitis, and sialalithiasis between September 2011 and January 2013, were retrospectively evaluated. The patients undergoing surgeries because of lymphadenopathy were selected among those scheduled for diagnostic excision. The patients with malign submandibular gland tumor or other suspected tumors at mouth base, those with a benign solid mass larger than 10 10 cm, and those who did not accept the transoral approach were excluded. All patients underwent neck sonography, whereas computed tomography scanning was performed in the patients with suspected malignity. All patients were informed that the procedure could be converted to a conventional wide-open transcervical approach in case of problematic conditions that cannot be accomplished via the transoral approach. All patients underwent surgery under general anesthesia by using nasotracheal intubation. The patients were placed in the supine position, with the neck semiextended and the head laterally rotated to the contralateral side. The surgeon stood at one side of the patients, and the assistant and scrub nurse stood at the left and right side of the patient. Endoscope (30-degree 4-mm rigid endoscope; Karl Storz Tuttlingen, Germany) was used in all patients, as it provides improved lightening, magnification of surgical field, and video capturing. The Digman mouth gag (Fixity Surgical Industries, Punjab, Pakistan) was placed into the oral cavity in an inverted fashion, and 2 tongue retractors were used to expose the mouth base. One percent lidocaine with a 1:100,000 dilution of epinephrine was injected to the mouth base mucosa. For the submandibular gland excision, a mucosal incision was made extending from the anterior of the Wharton canal to the
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retromolar trigone at approximately 1 cm medial to gingival, passing parallel to canal. At this point, sharp dissections were avoided to prevent injury to neurovascular structures, and blunt dissections were used. First, Wharton canal was identified and traced until the point where it crosses the lingual nerve. The lingual nerve was meticulously dissected by avoiding the deterioration of surgical exposure. Submandibular ganglion could be seen in some cases at the point where it branches from the lingual nerve; however, it might be disrupted in some cases as it is very fragile. The Wharton canal was divided by sparing the sublingual gland duct, and it was passed under the lingual nerve, which was then retracted posteriorly. Thus, a submandibular gland exposure was achieved that involved no nerve between the gland and canal. The submandibular gland is then progressively separated from the inferior border of the mylohyoid, the genioglossus, and the hyoglossus muscles by blunt dissections. At this point, the hypoglossal nerve could be identified in some cases, but it might be seen with a fascia-like structure overlying the nerve in a closed manner, and the hypoglossal nerve could be seen after dissection of this fascia-like structure in others. However, we did not observe the hypoglossal nerve as being in the gland or adherent to gland in any case including those with chronic sialadenitis. The facial artery and vein were identified at the posterolateral localization, which were clamped and secured, if indicated. However, the small branches of the facial artery and vein may rupture during blunt dissection, and intensive bleeding may occur for a while. These bleedings may spontaneously stop when a direct compression is applied by using gauze. After controlling the hemorrhage and neurovascular structures, surgical cavity was flushed by normal saline and closed via interrupted 4-0 rapid Vicryl sutures by leaving a small opening at the posterior for drainage. We also used the transoral lateral lingual approaches in the excision of dermoid cysts with a lateral localization, excision of submandibular lymphangioma, biopsies for lymphadenopathy (Fig. 1), and surgery of thyroglossal duct cysts localized at midline. However, as in submandibular gland excision, the Wharton canal, lingual nerve, hypoglossal nerve, gland, and digastric muscles were identified, and the target mass or cyst was excised by sparing these structures. In the excision of large masses localized under the mylohyoid muscle, the muscle is incised by a bipolar cautery device to facilitate dissection. In the midline surgeries, transfrenulum incision was made to excise dermoid cysts and thyroglossal duct cysts in particular. In case of dermoid cyst, the capsule of the cyst may be encountered after mucosal incision. The retraction of geniohyoid muscles may be needed in the dermoid cyst with more profound localization (Fig. 2). In case of thyroglossal duct cysts, dissection was advanced between genioglossus muscles after mucosal incision, and hyoid bone corpus was accessed by tracing the geniohyoid muscles. In the midline approach to a mass lesion, blunt dissection was preferred throughout the surgery after mucosal incision. Bone scissors was used at the point where the hyoid bone corpus was excised, whereas monopolar cautery device was used in the excision of the muscles adherent to hyoid bone.
FIGURE 2. In the dermoid cysts with midline and profound localization, it is difficult to ensure sufficient visualization despite retraction of genioglossus and geniohyoid muscles.
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RESULTS The mean age was 28.47 T 17.89 years (range, 8Y76 years) in 19 patients (9 men and 10 women) who were included (Table 1). Of these, submandibular gland excision was performed in 9 cases, whereas thyroglossal duct cyst excision was performed in 3 (2 transfrenulum approach and 1 lateral lingual approach), lymphangioma excision from submandibular region in 1, diagnostic lymph node excision at the posterior of submandibular gland in 3, and dermoid cyst excision (2 with midline and 1 with left submandibular localization) in 3 cases. Of the dermoid cysts, 2 were localized at submental region, whereas the other
Brief Clinical Studies
was localized at the left submandibular region. Mean surgery time was measured as 77.63 T 18.88 minutes (range, 45Y110 minutes). The shortest surgery time was recorded as 45 minutes in a midline dermoid cyst, whereas the longest surgery time was recorded as 110 minutes in a pleomorphic adenoma of the submandibular gland. The primary difficulty during surgery was dissection due to adhesion in the presence of limited exposure, which was particularly observed in 3 cases with chronic sialadenitis. Adhesions were observed at the anterior and anterolateral aspects of the gland in all submandibular gland excisions associated to difficulty in dissection due to adhesion. Of these, gland was excised in a fragmented manner
TABLE 1. Distribution of the Patients Who Underwent Mouth Base Surgeries by Transoral Approach in Terms of Clinical Diagnosis, Surgical Procedure, Perioperative Difficulties, Early Postoperative Complications, Pathological Results, and Surgery Time
No. Age,
Sex
Initial Diagnoses
1
9
Male
2
38
Female Right submandibular sialadenitis
3
76
Female Right submandibular sialadenitis
4
56
Female Left submandibular sialadenitis
5
39
Male
Left submandibular sialadenitis
6
12
Male
Left submandibular sialadenitis
7
30
Female Left submandibular sialadenitis
8
36
Female Right submandibular sialadenitis
9
35
Female Left submandibular gland adenoma 2.5 3 cm
10
19
11
31
12
32
13
45
14
10
15
22
16 17
8 16
Male Thyroglossal duct cyst Female Thyroglossal duct cyst
18 19
13 14
Female Thyroglossal duct cyst Male Left submandibular lymphangioma 6 9 cm
Left submandibular sialadenitis
Male
Surgical Approach Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland excision Transoral endoscopy-assisted gland and mass excision Transoral transfrenulum
Perioperative Difficulty and Complication
Early Postoperative Complications
None
None
Chronic sialadenitis
80
None
Chronic sialadenitis
90
Chronic sialadenitis with acute episodes
70
Chronic sialadenitis + sialalithiasis
90
Fragmented dissection of the gland due to adhesion Facial artery rupture
Difficult dissection of lingual nerve and Wharton duct due to adhesion Difficult dissection of gland due to adhesion
Edema at submandibular region and lingual ecchymosis Temporary abnormal tongue sensation
Left submandibular conglomerate lymphadenopathy Female Right submandibular lymphadenopathy
None
None
Surgery Time, min
100
None
None
Chronic sialadenitis
65
Fragmented dissection of the gland + facial artery rupture
None
Chronic sialadenitis
80
None
None
Chronic sialadenitis + sialalithiasis
65
None
Submandibular gland pleomorphic adenoma
None
Dermoid cyst
45
None
Dermoid cyst
50
None
Dermoid cyst
60
None
85
90 100 100 80
Rupture of adenoma capsule due to excessive size of mass
Submental dermoid Challenging dissection cyst 8 10 cm without causing rupture Female Submental dermoid Transoral transfrenulum None cyst 8 9 cm Male Left submandibular Transoral Elongation and dermoid cyst 5 8 cm endoscopy-assisted deformation of the duct cyst excision Female Left submandibular Transoral retroglandular Difficult exposure and dissection lymphadenopathy approach Male
Histopathology
Transoral retroglandular lymph node excision
None
None
Transoral retroglandular lymph node excision
None
None
Transoral transfrenulum Transoral lateral lingual approach Transoral transfrenulum Transoral lymphangioma and submandibular gland excision
Difficult exposure and dissection Difficult excision of hyoid bone Difficult exposure and dissection Difficult dissection inferior to mylohyoid muscle and rupture of the capsule
Submental edema None
Low-grade non-Hodgkin lymphoma Granulomatous lymphadenitis with necrosis Chronic granulomatous inflammation with caseification necrosis Thyroglossal duct cyst Thyroglossal duct cyst
Submental edema None
Thyroglossal duct cyst Lymphangioma
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60
55
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FIGURE 3. The gland can be removed in a fragmented manner in case of difficult dissection due to adhesion during submandibular gland excision.
in 2 cases (Fig. 3), whereas facial artery rupture occurred in another 2 cases, which were then ligated (Fig. 4). Capsule of the mass was ruptured in a case with submandibular adenoma, whereas dissection became more difficult because of rupture risk related to the extent of the cyst in dermoid cysts. A postoperative temporary abnormal tongue sensation was detected due to partial adhesion observed between Wharton canal and lingual nerve in a patient who underwent surgery for sialalithiasis (Fig. 5). Postoperative mouth base edema was detected in 2 cases with thyroglossal duct cyst, whereas postoperative mouth base edema and lingual ecchymosis were observed in a submandibular gland excision (Fig. 6). The cystic mass was localized inferior to the mylohyoid muscle in a case with lymphangioma, in which excision was performed via the transoral approach (Fig. 7). The exposure was improved by partial dissection of the muscle by using a bipolar electrocautery device (Fig. 8). The cyst rupture occurred despite this process. However, when ruptured cyst was evaluated, it was seen that cyst integrity was not disrupted, and en bloc cyst excision was possible. While visualization of surgical field and retraction of mouth base muscles were important problems in the thyroglossal duct cysts excised via midline approach (Fig. 9), resection of hyoid bone corpus was challenging in the lateral lingual approach (Fig. 10). A deformation was observed in the submandibular canal that resulted from the expansion of cystic mass and abnormal anatomical localization during the excision of dermoid cyst with lateral localization (Fig. 11).
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FIGURE 5. A partial adhesion is observed between Wharton canal and lingual nerve in a case with previous duct inflammation due to sialalithiasis (a: deformed Wharton duct containing a calculus, b: lingual nerve).
Partial synechia between Wharton canal and lingual nerve is one of the perioperative difficulties that can be observed in cases with sialadenitis, particularly in those with sialalithiasis. The nerve can be readily removed from duct by a meticulous dissection. Although the nerve does not rupture during this dissection, temporary abnormal tongue sensation or hypoesthesia may be observed. However, similar lingual paresthesia may be encountered in transcervical approaches in case of chronic sialalithiasis, if severe tissue adhesions are present.9,11 Weber et al2 found the rate of postoperative temporary abnormal tongue sensation as 43% in transoral surgical approaches, which were performed by leaving sufficient tissue in gingival mucosa to close the surgical field without excessive skeletonization of lingual nerve. In our case series where patients underwent similar surgical procedures, abnormal tongue sensation was observed in only 1 patient, which resolved at week 4 after surgery. In this case, a
mild adhesion was observed at the point where lingual nerve crosses the submandibular duct and dissection was hardly achieved. In cases with chronic sialadenitis, submandibular gland dissection can be rather difficult because of adhesion especially in the presence of previous abscess history. The adhesions were frequently observed in the anterolateral aspect of the gland, but no adhesion was observed in the base of gland or adjacent to the hypoglossal nerve. In these cases, the gland can be removed in a fragmented manner. In our study, the gland was excised in a fragmented manner in 2 cases. However, no gland tissue was left in the submandibular space. Again, difficulties in dissection did not cause paresthesia in the marginal mandibular branch of the facial nerve. Facial artery rupture is another perioperative complication that is rarely presented as a risk during blunt dissections. Facial artery rupture occurred in 2 cases in our study; however, bleeding was readily controlled by clamping the artery. Surgical clips can be used for this purpose; however, ligation of the facial artery in the transoral approach is not difficult for the surgeons performing classic tonsillectomy and preferring to ligate the tonsillary artery. Another concern is contamination or infection of the surgical field by oral flora. We use perioperative irrigation with chlorhexidine gluconate and administer antibiotic covering oral flora to reduce risk. However, there is a risk like this in cases undergoing tonsillectomy. Postoperative surgical infections are rarely observed in patients who underwent tonsillectomy; similarly, we observed no infection in patients who underwent surgeries via the transoral approach. There is a risk for perioperative paresthesia or paralysis of the marginal mandibular branch of the facial nerve, which is lower than that of transcervical approach, because it is localized in the subplatysmal plane, which protects it during transoral approach.1 Minimal invasive video-assisted sialoadenectomies are performed to avoid complications specific to transoral approach and scar tissue related to transcervical incision.3,12,13 However, temporary paresthesia of the marginal mandibular branch of facial nerve can be seen in these procedures as well.3 The transoral procedure performed in benign submandibular tumors such as adenoma is one of the factors that make perioperative surgical dissection difficult. In such cases, manipulation of a large mass in a limited area and en bloc dissection could be difficult. Moreover, integrity of the mass capsule can be disrupted. However, it is possible not to experience a recurrence if surgical field is sufficiently
FIGURE 4. Ruptured facial artery can be easily identified, which is then clamped and ligated (a: ruptured facial artery, b: hypoglossal nerve).
FIGURE 6. Lingual ecchymosis and edema at early postoperative period were seen in an elder patient with hypertension.
DISCUSSION
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FIGURE 7. Preoperative (A) and postoperative appearance (B) of a patient with lymphangioma excised by transoral route without an external incision.
eliminated without leaving residual tissue. Because it is known that the integrity of the capsule is occasionally disrupted during parotid gland surgery, no recurrence is observed unless any residual tissue is left, and the surgical field is sufficiently eliminated by irrigation. There is a rupture risk during dissection of cystic masses. The risk further increases in the transoral approach because of limited visualization and dissection space. We perform blunt dissection by placing angled clamps or angled part of a small retractor to the base of cyst especially in the dissection of the base. We did not observe any rupture even in the dermoid cysts up to 8 10 cm by using the previously mentioned process. The Sistrunk operation is the traditional surgical treatment in thyroglossal cysts, which is achieved by transcervical midline incision. Although the traditional approach is the only method in thyroglossal fistulas and in case of fistula formation between cyst and skin, the transoral approach can be preferred in case of undesirable skin incision or cysts without fistula formation. We observed diffuse edema that does not cause respiratory distress in patients who underwent surgery via the transoral approach. Alternatively, we used the lateral lingual approach in 1 case. We accessed the hyoid bone and cyst by tracing the digastric and hyoglossus muscles after exposing and sparing the submandibular gland and hypoglossal nerve. We observed less edema at the mouth base in this case. Another postoperative complication is edema at the mouth base and tongue, causing problems in mastication and swallowing after surgery. Increased edema due to trauma resulted from manipulation of muscles by retractors, and surgery may lead to scar tissue and contracture resulting in limitation of tongue movement in the long term. We make effort to not traumatize the muscles during surgery to reduce postoperative edema. We apply compression bandage to the submandibular or sublingual region at the end of surgery. In addition, we apply external cold by using cold packages and recommend drinking cold beverages. However, we did not observe edema at the tongue and mouth base sufficient to cause respiratory distress in any patient. The lateral approach is used in the submandibular gland excisions, and muscle dissection is limited in this approach other than retraction of mylohyoid muscle to anterolateral in a limited area. However, in the midline transfrenulum approach used in thyroglossal duct cysts, genioglossus and geniohyoid muscles are retracted by using retractors in the profound plane in particular. Thus, edema encountered is more massive in midline than in lateral approaches. Although midline approaches have the disadvantage of traumatizing muscles, it has the advantages such as lack of neurovascular structures. Another concern is laceration of the Wharton canal and caruncula in the surgery of thyroglossal duct and dermoid cysts via transoral midline incisions. This problem is not frequently observed
FIGURE 8. Mylohyoid muscle was partially dissected by bipolar electrocautery (A) and cystic mass was exposed (B).
Brief Clinical Studies
FIGURE 9. For transoral midline excisions of the thyroglossal cyst, the genioglossus muscles are retracted to lateral first, and the hyoid bone was accessed by tracing geniohyoid muscles, which has more profound localization (A) (a: genioglossus muscle, b: geniohyoid muscle, c: thyroglossal duct cyst). Preoperative (B) and postoperative appearance (C) of thyroglossal duct cyst without fistula formation between the cyst and skin.
in cases in which localization is achieved by the observation of saliva output through the caruncula after Wharton massage. Alternatively, this problem can be solved by initiating the first incision at a point over the frenulum close to the lingual end and advancing by blunt dissections close to caruncula. Dermoid cysts are commonly observed teratomatous congenital mass with benign features. Of all the dermoid cysts, 1% to 7% is detected in the head-neck region; of these, 23% are observed at the mouth base.14 The cyst is clinically classified into 3 types based on its relationship to muscles of the mouth base and the geniohyoid and mylohyoid muscles.15 The more common type is localized above the geniohyoid and mylohyoid muscles, which are clinically visible in the mouth base as they displace the tongue upward. If it is localized between the geniohyoid and the mylohyoid muscle or inferior to the mylohyoid muscle, it would appear as double chin. The third type is localized at an area where the cyst is displaced laterally into the submandibular area.16 In our clinic, we used excision via the transoral approach in all types. Total excision without disrupting capsule integrity is essential to avoid recurrences. In transoral approach, the problem is to achieve the excision in a limited area. We used blunt dissection throughout the surgery except the initial mucosal incision. Serious complications such as respiratory distress are seen in the untreated lymphangiomas localized at the neck and submandibular region.17 Sclerosing agents or surgical therapies can be used in the treatment of the lymphangiomas localized at this region.18 Recurrence has been reported in cases treated by injections of sclerosing agents.17 However, surgical therapies have undesirable outcomes such as incisions causing scar tissue. We excised a submandibular lymphangioma localized inferior to the mylohyoid muscles via the transoral approach. By this approach, cystic mass was totally resected without creating an incision scar at the skin. The successful removal of the lymphangioma despite difficult dissection due to limited visualization was a satisfactory outcome for patients. In our study, perioperative problems during diagnostic lymph node excision at the submandibular region included those related to identification of neurovascular structures and Wharton canal, which were observed in a similar appearance as in submandibular gland excision; in addition, it also included limited visualization and difficult dissection resulting from more posterior localization in retroglandular lymph node dissections. Endoscopic dissection is
FIGURE 10. Although more limited muscle retraction is performed in the lateral lingual approach to thyroglossal duct cyst, hyoid bone corpus can be barely resected, and it is warranted to expose neurovascular structures (a: hyoglossus muscle, b: lingual nerve, c: Wharton duct).
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Brief Clinical Studies
FIGURE 11. Deformation in submandibular canal and abnormal anatomical localization are seen in a dermoid cyst with lateral localization (A) (a: lingual nerve, b: duct of sublingual gland, c: common duct, d: Wharton duct). Neural and ductal structures are seen to be spared after the excision of the mass (B) (a: lingual nerve, b: Wharton duct, c: duct of sublingual gland).
helpful at this point. Early postoperative complications were similar to those seen in the submandibular gland excision. In conclusion, the transoral mouth base surgery is a safe surgical approach in selected patients. Permanent injuries of neural structures are rarely encountered, which is considered as a concern by head and neck surgeons. The hypoglossal nerve is located at the base of the submandibular gland; it is generally separated from the gland by a fascia-like soft tissue. Also, one cannot readily injure the hypoglossal nerve because of its thickness and durability, unless sharp dissection is used. Although the lingual nerve occasionally presents with adhesion to duct in cases with sialadenitis, no permanent injury is seen by meticulous dissection. Temporary complications and especially abnormal tongue sensations are well tolerated by the patients.
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Aneurysmal Bone Cyst of the Zygomatic Bone Jun Yong Lee, MD, Young-Il Ko, MD, Ho Kwon, MD, Sung-No Jung, MD Abstract: Aneurysmal bone cyst is a rare, non-neoplastic lesion that mostly involves the long bone and the spine, and is characterized by its expansile, vascular, and multi-cystic features. Reports of facial bone lesions are rare, and when it occurs, is usually located in the mandible. Herein, this report is aimed to describe a very rare case of an aneurysmal bone cyst in the zygoma with a brief review of the literature. Key Words: Aneurysmal bone cyst, zygomatic bone, neoplasm, bone tumor
A
neurysmal bone cyst is a septated pseudocyst that is devoid of epithelial lining and comprises a blood-filled cavity.1 It is commonly found in the long bone and the spine, and its incidence in the facial bone is rare, with most of these cases occurring in the mandible; 3 cases involving the zygoma have been reported to date.2 A 45-year-old female patient visited our outpatient department with a chief complaint of a left malar nodule that had developed about 15 months before presentation. There was no relevant history such as preceding facial trauma, and physical examination showed a nontender, hard, palpable mass on her left malar area. Computed tomography (CT) images revealed an intrabony, expansile lesion, with marked thinning of the surrounding zygoma (Fig. 1). No internal calcification within the mass was evident. Under general anesthesia, the authors completely excised the lesion through a subciliary incision; the resultant bone defect was repaired by filling in with bone putty (Allomatrix; Wright Medical Technology, Inc, Arlington, TN) (Fig. 2). Subsequent histopathologic examination showed a sinusoidal space filled with erythrocytes, fibroblasts, and histiocytes in a fibrous matrix (Fig. 3). No newly developed lesion was seen on a follow-up CT imaging performed 12 months postoperatively (Fig. 4), and the patient is currently on constant follow-up for 12 months without any signs of recurrence. From the Department of Plastic and Reconstructive Surgery, Uijeongbu St. Mary’s Hospital College of Medicine Catholic University of Korea, Seoul, Republic of Korea. Received August 08, 2013. Accepted for publication September 16, 2013. Address correspondence and reprint request to Sung-No Jung, M.D, Department of Plastic and Reconstructive Surgery, Uijeongbu St Mary’s Hospital, College of Medicine, Catholic University of Korea, 271 Cheonbo-ro, Uijeongbu-si, Gyeonggi-do, 480-717, Seoul, Republic of Korea; E-mail:[email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000463
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
