CLINICAL STUDY
Management of Carotid Body Tumor and Pseudoaneurysm After Blunt Dissection Wei-liang Chen, DDS, MD,* Lin-feng Xu, MD, PhD,† Qiong-lan Tang, MD, PhD,‡ and Da-ming Zhang, MS* Background: Surgical treatment of carotid body tumors remains challenging, and this study evaluated the outcomes of carotid body tumor and pseudoaneurysm after blunt dissection of the tumors. Methods: Six cases were classified as Shamblin groups I, II, and III (1, 1, and 4 cases, respectively). Tumor size ranged from 2 3 to 5 6 (median, 3.7 4.7) cm. Two patients underwent blunt dissection of the carotid body tumor, two underwent blunt dissection and ligation of the external carotid artery of the carotid body tumor, and two patients had common carotid artery–internal carotid artery artificial vascular reconstruction. Results: No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. Two patients had pseudoaneurysm or vocal cord paralysis postoperatively and recovered with stent graft implantation and medical treatment, respectively. The patients were followed for 6 to 17 (mean, 11) months, with no recurrence observed. Conclusion: Surgical treatment of a carotid body tumor is acceptably safe and effective according to Shamblin classification. Pseudoaneurysm can occur after blunt dissection of the tumor and can be treated with a stent graft. Key Words: carotid artery, carotid body tumor, pseudoaneurysm, reconstruction, Shamblin classification (J Craniofac Surg 2015;26: 477–480)
C
arotid body tumors are rare neoplasms arising from the paraganglion cells of the carotid body. They account for more than 50% of head and neck paragangliomas1; 96.7% are benign and 3.3% are malignant.2 Surgical treatment involves resection of the mass while maintaining the arterial structure if necessary, which can be challenging because the tumor is a highly vascular mass that is often densely adherent to the carotid bifurcation, and is exposed in a limited operative field. We report 6 cases of carotid body tumor and pseudoaneurysm after blunt dissection of the tumor.
From the *Departments of Oral and Maxillofacial Surgery, †Interventional Radiology, and ‡Pathology, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China. Received August 9, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Dr Wei-liang Chen, Department of Oral and Maxillofacial Surgery, Sun Yat-sen Memorial Hospital of Sun Yat-sen University, 107 Yan-jiang Road, 510120 Guangzhou, China; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001466
PATIENTS AND METHODS Between October 2011 and March 2013, 6 patients with carotid body tumors were admitted and treated at the Department of Oral and Maxillofacial Surgery, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China. Five patients were male and one was female. Their ages ranged from 16 to 63 (mean, 37.5) years. The main symptom that patients exhibited was a slowly growing mass with pulsation in the anterior cervical triangle region. No patient had neurological sequelae. Carotid body tumors were diagnosed based on patient histories, physical examinations, and imaging examinations, such as 3-dimensional computed tomography (3D-CT) and digital subtraction angiography. Preoperative Shamblin classification3 was performed, and the cases were assigned to Shamblin groups I (n = 1), II (n = 1), and III (n = 4). Tumor size ranged from 2 3 to 5 6 (median, 3.7 4.7) cm. The patients underwent surgical treatment under general anesthesia. Two underwent blunt dissection of the carotid body tumor, two underwent blunt dissection and ligation of the external carotid artery of the carotid body tumor, and two patients underwent common carotid artery–internal carotid artery artificial vascular reconstruction (Table 1). No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. All masses were confirmed pathologically as carotid body tumors with no trace of malignancy. No patient had hypoglossal or facial nerve pathology. Two patients had pseudoaneurysm or vocal cord paralysis postoperatively; 1 patient was treated with stent graft implantation and the other received medical treatment. The patients were followed for 6 to 17 (mean, 11) months, with no recurrence observed during the follow-up period (Table 1).
CLINICAL REPORTS Patient 1 A 63-year-old man presented with a mass on the right neck near the mandibular angle (Fig. 1). The 4 4 5-cm mass was slow growing, painless, and pulsatile. A 3D-CT image of the neck showed a large, well-defined, soft-tissue lesion just above the right carotid artery bifurcation that splayed the internal and external carotid arteries and encased the vessels almost completely (Fig. 2). The lesion was diagnosed as a Shamblin group III carotid body tumor. The patient underwent surgery under general anesthesia. The common carotid artery and branches were explored. The internal jugular vein and vagus and hypoglossal nerves were identified and separated from the surgical plane to prevent damage (Fig. 3). Before tumor resection, the external carotid artery and branches of the common carotid artery were dissected (Fig. 4). Dissection was attempted from the carotid bifurcation and extended onto the superior margin of the tumor, and the mass was separated completely from the common carotid artery and its branches in the subadventitial plane with Harmonic LigaSure (Fig. 5). At the time of tumor dissection, the quantity of blood loss was 750 mL, requiring no packed cell infusion. Pathological analysis identified the mass as a paraganglioma (Fig. 6). The patient was discharged on the seventh postoperative day without cranial nerve or vocal cord palsy.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Chen et al
TABLE 1. Diagnosis, Treatments, and Outcome of 6 Patients With Advanced Oral Cancer Case, Age (yr), Gender 1, 63, M 2, 16, M 3, 35, M 4, 32, F 5, 43, M 6, 36, M
Shamblin’s Classification
Size of Tumor (cm)
Treatment/Blood Loss (mL)
Complications
Follow-Up (mo)
Status
Type III Type III Type III Type III Type II Type I
45 56 45 45 34 23
BD + LEC/650 C-IAVR/550 C-IAVR/480 BD + LEC/500 BD/300 BD/250
Pseudoaneurysm Vocal cord paralysis No No No No
6 7 9 12 15 17
NR NR NR NR NR NR
M, male; F, female; BD, blunt dissection; BD + LEC, blunt dissection and ligation of external carotid artery; C-IAVR, common carotid artery–internal carotid artery artificial vascular reconstruction; NR, no recurrence.
One month postoperatively, the patient was referred to our department again with the complaints of pain and swelling in the right carotid artery region, difficulty swallowing, and dyspnea. Angiography showed a pseudoaneurysm arising from the right common carotid artery (Fig. 7), which was treated successfully with implantation of a Fluency Plus stent graft (Bard Peripheral Vascular, Tempe, AZ, USA) using an endovascular interventional technique (Fig. 8). Postoperative angiography demonstrated the absence of endoleakage. The patient recovered with no postoperative neurological symptom and no recurrence observed at a 6-month follow-up evaluation.
Patient 2
vocal cord medialization and medical treatment. He recovered within 3 months.
RESULTS No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. All masses were confirmed pathologically as carotid body tumors with no trace of malignancy. No patient had hypoglossal or facial nerve pathology. One patient with pseudoaneurysm and another with vocal cord paralysis postoperatively recovered after stent graft implantation and medical treatment, respectively. No artificial blood vessel or implanted stent graft was
A 16-year-old male patient presented with a mass on the right neck near the mandibular angle (Fig. 9). CT with angiography and 3D reconstruction showed a large, well-defined, soft-tissue lesion just above the right carotid artery bifurcation that splayed the internal and external carotid arteries and encased the vessels almost completely (Fig. 10). The mass was diagnosed as a Shamblin group III carotid body tumor. Under general anesthesia, the common carotid artery and branches were explored with Harmonic LigaSure. The internal jugular vein and vagus and hypoglossal nerves were identified and preserved. The common carotid artery and internal and external carotid arteries were excised along with the tumor, leaving a 0.5-cm margin of healthy tissue. After mass excision, a collagen-coated woven polyester vascular prosthesis (InterGard Woven, 8-cm length, 7-mm diameter; InterVascular S. A. S., La Ciotat, France) was implanted between the common and internal carotid arteries (Fig. 11). The quantity of intraoperative blood loss was 600 mL, and no packed cell infusion was required. Pathological analysis revealed that the mass was a paraganglioma (Fig. 12). Biopsy of 2 regional lymph nodes revealed no metastasis. The patient had no cerebrovascular sequela, but developed unilateral vocal cord paralysis on the lesion side and required
FIGURE 2. Three-dimensional computed tomographic image showing a 4.0 4.5-cm homogeneously and brightly enhancing right carotid body tumor that splays the internal and external carotid arteries.
FIGURE 1. A right carotid body tumor in a 63-year-old man.
FIGURE 3. The common carotid artery was explored and the internal jugular vein and vagus and hypoglossal nerves were dissected.
478
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 4. Ligation of the external carotid artery.
FIGURE 5. The carotid body tumor was resected with the external carotid artery. A, Intraoperative view after resection of the carotid body tumor with the external carotid artery. Photograph of carotid body tumor excision. B, En bloc tumor removal.
found to be occluded at 6 to 9 months after vascular reconstruction. The patients were followed for 6 to 17 (mean, 11) months, and no recurrence was observed during the follow-up period (Table 1).
Management of Carotid Body Tumor
FIGURE 7. Lateral view of the right common carotid artery. Digital subtraction angiography revealed a 4 4-cm pseudoaneurysm arising from the right common carotid artery.
reconstruction. Two patients had vocal cord paralysis postoperatively, but all survived. A Shamblin group I tumor partially encases the vessels and can be easily dissected from the vessels with no adventitial infiltration. The dimensions and pathological characteristics of Shamblin I and II tumors allow dissection, which was attempted in our case from the arterial bifurcation and extended onto the superior margin of the tumor. The tumor was dissected completely from the common carotid artery and its branches in the subadventitial plane.8,9 Rodríguez-Cuevas et al2 reported that complete resection is achieved in 85% of cases. Shamblin group III tumors encase the vessels almost completely. Of the 4 patients with Shamblin III tumors in our series, 2 had no obvious adventitial infiltration and underwent blunt dissection and ligation of the external carotid artery of the tumor. One patient had a pseudoaneurysm that required stent graft implantation. The other 2
DISCUSSION Surgical resection is the recommended treatment for carotid body tumor. As the excision of this tumor type involves high risks of cranial nerve paresis and carotid artery injury, careful handling and good surgical skills are required to ensure complete removal.4 The postsurgical mortality rate is reported to be 5%–13%, the postoperative cerebrovascular insufficiency rate is 8%–20%,5,6 and the postoperative cranial nerve paralysis rate is 32%–44%.7 Sanli et al.8 reported 1 death and 2 cases of vocal cord paralysis among 8 patients with Shamblin I and II tumors treated with blunt dissection and 1 patient with a Shamblin III tumor who underwent mass resection and synthetic polytetrafluoroethylene graft implantation. In our series, 2 patients with Shamblin I and II tumors were treated with blunt dissection by thermal cautery in the subadventitial plane; 2 patients with Shamblin III tumors underwent blunt dissection and ligation of the external carotid artery of the tumor, and the other two Shamblin III cases, which involved tumor invasion of the carotid artery and adjacent tissues, underwent mass resection and artificial vascular
FIGURE 6. Nested arrangement of tumor cells (H&E, original magnification 100).
FIGURE 8. Lateral view of the right common carotid artery and endovascular repair of the carotid pseudoaneurysm with implantation of an 8 40-mm-long Fluency Plus stent graft.
FIGURE 9. A right carotid body tumor in a 16-year-old male patient.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
479
Chen et al
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 12. The arterial wall is infiltrated. FIGURE 10. Computed tomography with angiography and 3-dimensional reconstruction showing a 5 6-cm enhancing right carotid body tumor that splays the internal and external carotid arteries.
Shamblin III tumors, which were difficult to dissect from the vessels and had partial or focal adventitial infiltration, required resection of the external and internal carotid arteries and artificial collagen-coated woven polyester vascular prosthesis implantation. Arterial continuity was maintained through graft interposition in these cases. The selection of an operating team with experience in vascular reconstructive techniques is strongly recommended to reduce the risk of major vascular injury, especially in Shamblin class III tumors.4 Postoperative complications of this procedure include stroke, transient cranial nerve deficits, permanent Horner syndrome, and cerebrospinal fluid leakage.10 We were unable to locate any report of pseudoaneurysm development after blunt dissection of a carotid body tumor. Pseudoaneurysm is usually caused by blunt injury or trauma, such as carotid endarterectomy.11 It tends to enlarge progressively and compress the surrounding structures, causing symptoms such as head and neck pain, dysphagia, and dyspnea. This lesion can eventually rupture and is potentially fatal. The standard of treatment for carotid pseudoaneurysm is open surgical repair with excision of the defect, followed by arterial reconstruction with a graft. To our knowledge, this report is the first to describe the occurrence of carotid pseudoaneurysm after blunt dissection of a carotid body tumor, which was successfully treated by implantation of an 8 40-mm-long Fluency Plus stent graft to seal the leakage from the common carotid artery to the pseudoaneurysm. Endovascular interventional techniques can be used to treat carotid pseudoaneurysm using a stent graft.12 Paragangliomas are highly vascular tumors of neural crest origin that involve the walls of blood vessels or specific nerves in the head and neck. For large carotid body tumors, preoperative embolization may simplify the operation and reduce blood loss.13 The use of Harmonic LigaSure devices resulted in a mean blood loss of 455 (range, 250–650) mL in our series. LigaSure is an electrothermal
FIGURE 11. The common carotid artery was reconstructed with an InterGard Woven collagen-coated woven polyester vascular prosthesis. A, Intraoperative view. B, Three-dimensional computed tomography angiographic image showing the artificial blood vessel.
480
sealing system that seals the vessel through physical pressure and bipolar electrical coagulation, reducing thermal spread. Some authors have reported that the use of LigaSure in carotid body tumor surgery reduced average blood loss (by 285 mL) and operative time.14 In conclusion, the surgical treatment of Shamblin carotid body tumors is safe and effective. Shamblin I and II tumors can be treated with blunt dissection, and Shamblin III tumors can be treated with mass resection and synthetic polytetrafluoroethylene graft implantation. Pseudoaneurysm can occur after blunt dissection of the tumor, and stent grafts are feasible for the treatment of this lesion. Harmonic LigaSure devices may reduce intraoperative blood loss.
REFERENCES 1. Rao AB, Koeller KK, Adair CF. From the archives of the AFIP. Paragangliomas of the head and neck: radiologic-pathologic correlation. Armed Forces Institute of Pathology. Radiographics 1999;19:1605–1632 2. Rodríguez-Cuevas S, López-Garza J, Labastida-Almendaro S. Carotid body tumors in inhabitants of altitudes higher than 2000 meters above sea level. Head Neck 1998;20:374–378 3. Shamblin WR, ReMine WH, Sheps SG, et al. Carotid body tumor (chemodectoma). Clinicopathologic analysis of ninety cases. Am J Surg 1971;122:732–739 4. Erdogan BA, Bora F, Altin G, et al. Our experience with carotid body paragangliomas. Prague Med Rep 2012;113:262–270 5. Hirsch JH, Killien FC, Troupin RH. Bilateral carotid body tumors and cyanotic heart disease. AJR Am J Roentgenol 1980;134:1073–1075 6. Keating JF, Miller GA, Keaveny TV. Carotid body tumours: report of six cases and a review of management. J R Coll Surg Edinb 1990;35:172–174 7. Persky MS, Setton A, Niimi Y, et al. Combined endovascular and surgical treatment of head and neck paragangliomas—a team approach. Head Neck 2002;24:423–431 8. Sanli A, Oz K, Ayduran E, et al. Carotid body tumors and our surgical approaches. Indian J Otolaryngol Head Neck Surg 2012;64:158–161 9. Arya S, Rao V, Juvekar S, et al. Carotid body tumors: objective criteria to predict the Shamblin group on MR imaging. AJNR Am J Neuroradiol 2008;29:1349–1354 10. Karaman E, Isildak H, Yilmaz M, et al. Management of paragangliomas in otolaryngology practice: review of a 7-year experience. J Craniofac Surg 2009;20:1294–1297 11. Troutman DA, Mohan CR, Samhouri FA, et al. Endovascular repair of carotid artery pseudoaneurysm after carotid endarterectomy with self-expanding covered stents—a long-term follow-up. Ann Vasc Surg 2010;24:954.e13–954.e16 12. Dahya V, Chalasani P. Successful obliteration of a pseudoaneurysm from post-CEA repair secondary to a Pruitt-Inahara shunt using a stent graft. Case Rep Vasc Med 2013;2013:382485 doi: 10.1155/2013/382485. Epub 2013 Jul 31 13. Power AH, Bower TC, Kasperbauer J, et al. Impact of preoperative embolization on outcomes of carotid body tumor resections. J Vasc Surg 2012;56:979–989 14. Luna-Ortiz K, Villavicencio-Valencia V, Carmona-Luna T, et al. Carotid body tumour resection with LigaSure device. Acta Otorrinolaringol Esp 2010;61:6–11
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Management of Carotid Body Tumor and Pseudoaneurysm After Blunt Dissection Wei-liang Chen, DDS, MD,* Lin-feng Xu, MD, PhD,† Qiong-lan Tang, MD, PhD,‡ and Da-ming Zhang, MS* Background: Surgical treatment of carotid body tumors remains challenging, and this study evaluated the outcomes of carotid body tumor and pseudoaneurysm after blunt dissection of the tumors. Methods: Six cases were classified as Shamblin groups I, II, and III (1, 1, and 4 cases, respectively). Tumor size ranged from 2 3 to 5 6 (median, 3.7 4.7) cm. Two patients underwent blunt dissection of the carotid body tumor, two underwent blunt dissection and ligation of the external carotid artery of the carotid body tumor, and two patients had common carotid artery–internal carotid artery artificial vascular reconstruction. Results: No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. Two patients had pseudoaneurysm or vocal cord paralysis postoperatively and recovered with stent graft implantation and medical treatment, respectively. The patients were followed for 6 to 17 (mean, 11) months, with no recurrence observed. Conclusion: Surgical treatment of a carotid body tumor is acceptably safe and effective according to Shamblin classification. Pseudoaneurysm can occur after blunt dissection of the tumor and can be treated with a stent graft. Key Words: carotid artery, carotid body tumor, pseudoaneurysm, reconstruction, Shamblin classification (J Craniofac Surg 2015;26: 477–480)
C
arotid body tumors are rare neoplasms arising from the paraganglion cells of the carotid body. They account for more than 50% of head and neck paragangliomas1; 96.7% are benign and 3.3% are malignant.2 Surgical treatment involves resection of the mass while maintaining the arterial structure if necessary, which can be challenging because the tumor is a highly vascular mass that is often densely adherent to the carotid bifurcation, and is exposed in a limited operative field. We report 6 cases of carotid body tumor and pseudoaneurysm after blunt dissection of the tumor.
From the *Departments of Oral and Maxillofacial Surgery, †Interventional Radiology, and ‡Pathology, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China. Received August 9, 2014. Accepted for publication December 1, 2014. Address correspondence and reprint requests to Dr Wei-liang Chen, Department of Oral and Maxillofacial Surgery, Sun Yat-sen Memorial Hospital of Sun Yat-sen University, 107 Yan-jiang Road, 510120 Guangzhou, China; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001466
PATIENTS AND METHODS Between October 2011 and March 2013, 6 patients with carotid body tumors were admitted and treated at the Department of Oral and Maxillofacial Surgery, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China. Five patients were male and one was female. Their ages ranged from 16 to 63 (mean, 37.5) years. The main symptom that patients exhibited was a slowly growing mass with pulsation in the anterior cervical triangle region. No patient had neurological sequelae. Carotid body tumors were diagnosed based on patient histories, physical examinations, and imaging examinations, such as 3-dimensional computed tomography (3D-CT) and digital subtraction angiography. Preoperative Shamblin classification3 was performed, and the cases were assigned to Shamblin groups I (n = 1), II (n = 1), and III (n = 4). Tumor size ranged from 2 3 to 5 6 (median, 3.7 4.7) cm. The patients underwent surgical treatment under general anesthesia. Two underwent blunt dissection of the carotid body tumor, two underwent blunt dissection and ligation of the external carotid artery of the carotid body tumor, and two patients underwent common carotid artery–internal carotid artery artificial vascular reconstruction (Table 1). No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. All masses were confirmed pathologically as carotid body tumors with no trace of malignancy. No patient had hypoglossal or facial nerve pathology. Two patients had pseudoaneurysm or vocal cord paralysis postoperatively; 1 patient was treated with stent graft implantation and the other received medical treatment. The patients were followed for 6 to 17 (mean, 11) months, with no recurrence observed during the follow-up period (Table 1).
CLINICAL REPORTS Patient 1 A 63-year-old man presented with a mass on the right neck near the mandibular angle (Fig. 1). The 4 4 5-cm mass was slow growing, painless, and pulsatile. A 3D-CT image of the neck showed a large, well-defined, soft-tissue lesion just above the right carotid artery bifurcation that splayed the internal and external carotid arteries and encased the vessels almost completely (Fig. 2). The lesion was diagnosed as a Shamblin group III carotid body tumor. The patient underwent surgery under general anesthesia. The common carotid artery and branches were explored. The internal jugular vein and vagus and hypoglossal nerves were identified and separated from the surgical plane to prevent damage (Fig. 3). Before tumor resection, the external carotid artery and branches of the common carotid artery were dissected (Fig. 4). Dissection was attempted from the carotid bifurcation and extended onto the superior margin of the tumor, and the mass was separated completely from the common carotid artery and its branches in the subadventitial plane with Harmonic LigaSure (Fig. 5). At the time of tumor dissection, the quantity of blood loss was 750 mL, requiring no packed cell infusion. Pathological analysis identified the mass as a paraganglioma (Fig. 6). The patient was discharged on the seventh postoperative day without cranial nerve or vocal cord palsy.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
477
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
Chen et al
TABLE 1. Diagnosis, Treatments, and Outcome of 6 Patients With Advanced Oral Cancer Case, Age (yr), Gender 1, 63, M 2, 16, M 3, 35, M 4, 32, F 5, 43, M 6, 36, M
Shamblin’s Classification
Size of Tumor (cm)
Treatment/Blood Loss (mL)
Complications
Follow-Up (mo)
Status
Type III Type III Type III Type III Type II Type I
45 56 45 45 34 23
BD + LEC/650 C-IAVR/550 C-IAVR/480 BD + LEC/500 BD/300 BD/250
Pseudoaneurysm Vocal cord paralysis No No No No
6 7 9 12 15 17
NR NR NR NR NR NR
M, male; F, female; BD, blunt dissection; BD + LEC, blunt dissection and ligation of external carotid artery; C-IAVR, common carotid artery–internal carotid artery artificial vascular reconstruction; NR, no recurrence.
One month postoperatively, the patient was referred to our department again with the complaints of pain and swelling in the right carotid artery region, difficulty swallowing, and dyspnea. Angiography showed a pseudoaneurysm arising from the right common carotid artery (Fig. 7), which was treated successfully with implantation of a Fluency Plus stent graft (Bard Peripheral Vascular, Tempe, AZ, USA) using an endovascular interventional technique (Fig. 8). Postoperative angiography demonstrated the absence of endoleakage. The patient recovered with no postoperative neurological symptom and no recurrence observed at a 6-month follow-up evaluation.
Patient 2
vocal cord medialization and medical treatment. He recovered within 3 months.
RESULTS No perioperative mortality or stroke occurred. The mean blood loss was 455 (range, 250–650) mL. All masses were confirmed pathologically as carotid body tumors with no trace of malignancy. No patient had hypoglossal or facial nerve pathology. One patient with pseudoaneurysm and another with vocal cord paralysis postoperatively recovered after stent graft implantation and medical treatment, respectively. No artificial blood vessel or implanted stent graft was
A 16-year-old male patient presented with a mass on the right neck near the mandibular angle (Fig. 9). CT with angiography and 3D reconstruction showed a large, well-defined, soft-tissue lesion just above the right carotid artery bifurcation that splayed the internal and external carotid arteries and encased the vessels almost completely (Fig. 10). The mass was diagnosed as a Shamblin group III carotid body tumor. Under general anesthesia, the common carotid artery and branches were explored with Harmonic LigaSure. The internal jugular vein and vagus and hypoglossal nerves were identified and preserved. The common carotid artery and internal and external carotid arteries were excised along with the tumor, leaving a 0.5-cm margin of healthy tissue. After mass excision, a collagen-coated woven polyester vascular prosthesis (InterGard Woven, 8-cm length, 7-mm diameter; InterVascular S. A. S., La Ciotat, France) was implanted between the common and internal carotid arteries (Fig. 11). The quantity of intraoperative blood loss was 600 mL, and no packed cell infusion was required. Pathological analysis revealed that the mass was a paraganglioma (Fig. 12). Biopsy of 2 regional lymph nodes revealed no metastasis. The patient had no cerebrovascular sequela, but developed unilateral vocal cord paralysis on the lesion side and required
FIGURE 2. Three-dimensional computed tomographic image showing a 4.0 4.5-cm homogeneously and brightly enhancing right carotid body tumor that splays the internal and external carotid arteries.
FIGURE 1. A right carotid body tumor in a 63-year-old man.
FIGURE 3. The common carotid artery was explored and the internal jugular vein and vagus and hypoglossal nerves were dissected.
478
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 4. Ligation of the external carotid artery.
FIGURE 5. The carotid body tumor was resected with the external carotid artery. A, Intraoperative view after resection of the carotid body tumor with the external carotid artery. Photograph of carotid body tumor excision. B, En bloc tumor removal.
found to be occluded at 6 to 9 months after vascular reconstruction. The patients were followed for 6 to 17 (mean, 11) months, and no recurrence was observed during the follow-up period (Table 1).
Management of Carotid Body Tumor
FIGURE 7. Lateral view of the right common carotid artery. Digital subtraction angiography revealed a 4 4-cm pseudoaneurysm arising from the right common carotid artery.
reconstruction. Two patients had vocal cord paralysis postoperatively, but all survived. A Shamblin group I tumor partially encases the vessels and can be easily dissected from the vessels with no adventitial infiltration. The dimensions and pathological characteristics of Shamblin I and II tumors allow dissection, which was attempted in our case from the arterial bifurcation and extended onto the superior margin of the tumor. The tumor was dissected completely from the common carotid artery and its branches in the subadventitial plane.8,9 Rodríguez-Cuevas et al2 reported that complete resection is achieved in 85% of cases. Shamblin group III tumors encase the vessels almost completely. Of the 4 patients with Shamblin III tumors in our series, 2 had no obvious adventitial infiltration and underwent blunt dissection and ligation of the external carotid artery of the tumor. One patient had a pseudoaneurysm that required stent graft implantation. The other 2
DISCUSSION Surgical resection is the recommended treatment for carotid body tumor. As the excision of this tumor type involves high risks of cranial nerve paresis and carotid artery injury, careful handling and good surgical skills are required to ensure complete removal.4 The postsurgical mortality rate is reported to be 5%–13%, the postoperative cerebrovascular insufficiency rate is 8%–20%,5,6 and the postoperative cranial nerve paralysis rate is 32%–44%.7 Sanli et al.8 reported 1 death and 2 cases of vocal cord paralysis among 8 patients with Shamblin I and II tumors treated with blunt dissection and 1 patient with a Shamblin III tumor who underwent mass resection and synthetic polytetrafluoroethylene graft implantation. In our series, 2 patients with Shamblin I and II tumors were treated with blunt dissection by thermal cautery in the subadventitial plane; 2 patients with Shamblin III tumors underwent blunt dissection and ligation of the external carotid artery of the tumor, and the other two Shamblin III cases, which involved tumor invasion of the carotid artery and adjacent tissues, underwent mass resection and artificial vascular
FIGURE 6. Nested arrangement of tumor cells (H&E, original magnification 100).
FIGURE 8. Lateral view of the right common carotid artery and endovascular repair of the carotid pseudoaneurysm with implantation of an 8 40-mm-long Fluency Plus stent graft.
FIGURE 9. A right carotid body tumor in a 16-year-old male patient.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
479
Chen et al
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 12. The arterial wall is infiltrated. FIGURE 10. Computed tomography with angiography and 3-dimensional reconstruction showing a 5 6-cm enhancing right carotid body tumor that splays the internal and external carotid arteries.
Shamblin III tumors, which were difficult to dissect from the vessels and had partial or focal adventitial infiltration, required resection of the external and internal carotid arteries and artificial collagen-coated woven polyester vascular prosthesis implantation. Arterial continuity was maintained through graft interposition in these cases. The selection of an operating team with experience in vascular reconstructive techniques is strongly recommended to reduce the risk of major vascular injury, especially in Shamblin class III tumors.4 Postoperative complications of this procedure include stroke, transient cranial nerve deficits, permanent Horner syndrome, and cerebrospinal fluid leakage.10 We were unable to locate any report of pseudoaneurysm development after blunt dissection of a carotid body tumor. Pseudoaneurysm is usually caused by blunt injury or trauma, such as carotid endarterectomy.11 It tends to enlarge progressively and compress the surrounding structures, causing symptoms such as head and neck pain, dysphagia, and dyspnea. This lesion can eventually rupture and is potentially fatal. The standard of treatment for carotid pseudoaneurysm is open surgical repair with excision of the defect, followed by arterial reconstruction with a graft. To our knowledge, this report is the first to describe the occurrence of carotid pseudoaneurysm after blunt dissection of a carotid body tumor, which was successfully treated by implantation of an 8 40-mm-long Fluency Plus stent graft to seal the leakage from the common carotid artery to the pseudoaneurysm. Endovascular interventional techniques can be used to treat carotid pseudoaneurysm using a stent graft.12 Paragangliomas are highly vascular tumors of neural crest origin that involve the walls of blood vessels or specific nerves in the head and neck. For large carotid body tumors, preoperative embolization may simplify the operation and reduce blood loss.13 The use of Harmonic LigaSure devices resulted in a mean blood loss of 455 (range, 250–650) mL in our series. LigaSure is an electrothermal
FIGURE 11. The common carotid artery was reconstructed with an InterGard Woven collagen-coated woven polyester vascular prosthesis. A, Intraoperative view. B, Three-dimensional computed tomography angiographic image showing the artificial blood vessel.
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sealing system that seals the vessel through physical pressure and bipolar electrical coagulation, reducing thermal spread. Some authors have reported that the use of LigaSure in carotid body tumor surgery reduced average blood loss (by 285 mL) and operative time.14 In conclusion, the surgical treatment of Shamblin carotid body tumors is safe and effective. Shamblin I and II tumors can be treated with blunt dissection, and Shamblin III tumors can be treated with mass resection and synthetic polytetrafluoroethylene graft implantation. Pseudoaneurysm can occur after blunt dissection of the tumor, and stent grafts are feasible for the treatment of this lesion. Harmonic LigaSure devices may reduce intraoperative blood loss.
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© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
