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First-bite syndrome: A review of 29 patients with parapharyngeal space tumor
Auris Nasus Larynx 35 (2008) 109–113 www.elsevier.com/locate/anl
First-bite syndrome: A review of 29 patients with parapharyngeal space tumor Yoshiyuki Kawashima a,*, Takuro Sumi a, Taro Sugimoto a, Seiji Kishimoto b a
Department of Otolaryngology, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan b Department of Head and Neck Surgery, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan Received 25 February 2007; accepted 25 June 2007 Available online 11 September 2007
Abstract Objective: The aim of this study is to clarify the etiology of first-bite syndrome. Methods: A total of 29 consecutive patients treated with a surgical resection for parapharyngeal space tumor between April 1999 and April 2005 were retrospectively reviewed. Results: Nine cases of first-bite syndrome were developed. Eight of those patients had undergone surgical ablation of the cervical sympathetic chain and/or external carotid artery. In two patients who underwent ablation of the sympathetic chain but in whom first-bite syndrome did not develop, the superior cervical ganglion was probably preserved. Conclusion: The present findings strongly indicate that loss of sympathetic innervation to the parotid gland from the superior cervical ganglion causes first-bite syndrome. They also suggest that some residual or autonomous activity of the superior cervical ganglion could prevent development of first-bite syndrome even if the lower sympathetic trunk was ablated. # 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: First-bite syndrome; Superior cervical ganglion; Parapharyngeal space; Complications
1. Introduction In 1986, Haubrich [1] first described first-bite syndrome (FBS). Typically, patients report excruciating pain in the parotid region only after the first bite of a meal. The pain improves with the subsequent masticatory movements of that meal, but invariably returns to the excruciating level at the first bite of the next meal [2]. The management of this syndrome can be troublesome. Treatment usually involves nonsteroidal anti-inflammatory medications or even carbamazepine, both of which provide poor results; all attempts at surgical treatment have been unsuccessful [2]. The pathogenesis of FBS is largely unknown, but researchers have proposed that it involves loss of sympathetic nerve innervation to the parotid gland after operations involving * Corresponding author. Tel.: +81 3 5803 5308; fax: +81 3 3813 2134. E-mail address: [email protected] (Y. Kawashima).
the parapharyngeal space (PPS), causing denervation supersensitivity of salivary gland myoepithelial cells [3]. We noticed that not all patients who underwent surgical ablation of the cervical sympathetic chain developed FBS, and hypothesized that patients who underwent surgical ablation of the cervical sympathetic chain but did not develop FBS could provide the key to understand the etiology of FBS. In the present study, we reviewed our patients who underwent PPS resection with or without subsequent occurrence of FBS, and reviewed the relevant literature.
2. Materials and methods We retrospectively reviewed the medical records of all patients who underwent surgery to remove a PPS tumor at Tokyo Medical and Dental University Hospital between
0385-8146/$ – see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.anl.2007.06.005
110
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
April 1999 and April 2005. The medical records were reviewed for the following information: patient demographics (age, sex); clinical manifestations; radiographic findings; methods of treatment, including surgical notes; histological reports and complications. To differentiate FBS from other sources of pain, we excluded from analysis all patients who had temporomandibular joint pain postoperatively or who were diagnosed with malignant tumors.
3. Results Twenty-nine patients with a PPS tumor were treated over a 7-year period (1999–2005). Of these 29 tumors, 6 (21%) were found to be malignant. Another patient had experienced severe trismus, temporomandibular joint pain, and dysphagia postoperatively. Thus, 7 of the 29 cases were excluded from the study, leaving 22 cases. The mean age of these 22 patients (10 men and 12 women) at the time of surgery was 40 years (range, 10–66 years). For each of the 22 patients, Table 1 shows whether FBS developed, its site of
origin, histology, size, surgical approach, ablated structures, and complications. Ten patients (45%) had pleomorphic adenomas arising from the deep lobe of the parotid gland or minor salivary gland; in 4 of these 10 patients, the adenoma was continuous with the deep lobe of the parotid gland. The remaining 12 cases were as follows: five patients (23%) had sympathetic chain schwannomas; two patients (9%) had vagus nerve schwannomas; one patient had an auriculotemporal nerve schwannoma; two patients (9%) had carotid body tumors (paragangliomas); one patient had an oncocytoma; and one patient had a lipoma. The mean major axis length of the tumors was 5.9 cm (range, 3–9 cm). Various surgical approaches were used to resect the tumors. A transcervical approach was used to resect 11 tumors: four sympathetic schwannomas, two vagus nerve schwannomas, two carotid body tumors, two pleomorphic adenomas, and one lipoma. A transcervical–transparotid approach was used to resect nine tumors: seven pleomorphic adenomas, one sympathetic nerve schwannoma, and one oncocytoma. A transcervical–transparotid approach with mandibulotomy was used to resect one auriculotemporal nerve schwannoma.
Table 1 Diagnosis, clinical features, and histological features
FBS, first-bite syndrome; C, transcervical; CP, transcervical–transparotid; CO, transcervical–transoral; M, mandibulotomy.
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
A transcervical–transoral approach was used to resect one pleomorphic adenoma. FBS developed in nine patients (41%; three men and six women), who had a median age of 42 years (range, 17–66 years). The mean major axis length of those nine tumors was 5.9 cm (range, 4–8 cm). A transcervical–transparotid approach was used to resect six of those tumors; a transcervical approach was used to resect the other three tumors. All nine patients had undergone surgical resection of structures within the PPS ipsilateral to the symptomatic side. None of the nine patients had a preoperative finding of FBS, Horner’s syndrome, or cranial nerve deficits. Three of the nine patients had sympathetic chain schwannoma. All three of those patients had undergone ablation of the cervical sympathetic chain and developed Horner’s syndrome postoperatively. The other six patients had pleomorphic adenoma arising from the deep lobe of the parotid gland or minor salivary gland; three of these six tumors were continuous with the deep lobe of the parotid gland. Patients with salivary gland tumors (cases 4–8) underwent ligation of the external carotid artery proximal to the deep surface of the parotid gland, and all five of those patients developed FBS. One patient (case 9) who underwent resection of the deep lobe of the parotid gland and did not undergo ligation of the external carotid artery also developed FBS. Two patients (cases 10 and 11) who underwent resection of a sympathetic chain schwannoma did not develop FBS. One patient (case 12) with an auriculotemporal nerve schwannoma underwent ligation of his external carotid artery, but did not develop FBS. Fig. 1 shows the relationships between development of FBS and ablated structures. The first thing we noticed is that in patients who underwent surgical ablation of the cervical sympathetic chain, FBS did not necessarily develop. With the exception of patient 12, FBS developed in patients who
Fig. 1. Relationships between development of first-bite syndrome and ablated structures. First-bite syndrome did not develop in all patients who underwent surgical ablation of the cervical sympathetic chain. With the exception of one patient (case 12), first-bite syndrome developed in patients who had undergone ligation of the external carotid artery.
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underwent ligation of the external carotid artery. With the exception of patient 9, FBS did not develop in patients who did not undergo ablation of the sympathetic chain and/or external carotid artery.
4. Comment In 1986, Haubrich [1] first described FBS. Since 1998, when Netterville et al. [3] described FBS caused by PPS resection, FBS has been well known to head and neck surgeons. FBS is the most common of the various surgical complications of PPS surgery that have been reported in the literature [4]. The pain of FBS is due to severe cramping or spasm in the parotid gland region at the first bite of each meal; the pain diminishes over the next several bites [2]. The cause of FBS is largely unknown, but Netterville et al. [3] have proposed that FBS is caused by the loss of sympathetic innervation to the parotid gland. Before discussing the possible mechanisms of postoperative FBS, we should review the autonomous innervation of the parotid gland (Fig. 2). The parotid gland is innervated by both sympathetic and parasympathetic pathways. The sympathetic pathway can be regarded as a threeneuron pathway. The first-order neurons of the sympathetic pathway originate in the central nervous system (in the hypothalamus), and descend through the brainstem to a synapse in the thoracic region of the spinal cord. The second-order neurons exit the spinal cord at the first thoracic segment, and enter the sympathetic chain ascending through the inferior and middle cervical ganglia, synapsing in the superior cervical ganglion. The superior cervical ganglion is roughly 2.5 cm long and is located at the level of the second and third cervical vertebrae, posterior to the carotid sheath, between the internal carotid artery and the longus capitis muscle [5]. Anterior branches are distributed along the external and internal carotid arteries. Postganglionic nerves that innervate the eyeball, eyelid or orbit run in the internal carotid nerve and plexus. Most of the fibers that innervate the sweat glands or skin of the face or scalp run along the external carotid artery. The sympathetic nerve supply to the parotid gland itself branches from the external carotid artery plexus to travel along the middle meningeal artery. Thus, a loss of sympathetic innervation to the parotid gland can occur even in the absence of Horner’s syndrome. The parasympathetic innervation of the parotid gland consists mainly of ninth nerve impulses originating in the inferior salivary nucleus, which arrive at the otic ganglion via the glossopharyngeal nerve and tympanic plexus. From the otic ganglion, postganglionic fibers to the parotid gland pass to the neighboring auriculotemporal branch of the mandibular nerve. Netterville et al. [3] reviewed 46 vagal paragangliomas. In their series, nine patients exhibited FBS, and all nine of those patients had undergone resection of their sympathetic chain and/or exhibited loss of sympathetic function
112
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
Fig. 2. Pathway of autonomous nervous supply to the parotid gland.
manifested by Horner’s syndrome [3]. Two other patients underwent resection of the sympathetic trunk, and did not report parotid pain. Based on these findings, Netterville et al. [3] proposed that FBS was caused by damage or removal of the cervical sympathetic trunk with loss of sympathetic innervation to the parotid gland. In 2002, to assess the anatomical basis of this proposal, Chiu et al. [2] reviewed the records of 12 patients with FBS. Six patients exhibited postoperative Horner’s syndrome. The other six patients had undergone external carotid artery ligation inferior to the parotid gland. These two studies strongly suggest that FBS is caused by loss of sympathetic innervation of the parotid gland. Although most of the present cases strongly support the proposal of Netterville et al. [3], there are some exceptions (Fig. 1). One patient (case 12) who underwent ligation of the external carotid artery did not develop FBS. This patient, who had an auriculotemporal nerve schwannoma, underwent ligation of the auriculotemporal nerve as well as the external carotid artery. The reason why FBS did not develop in that patient is probably that both sympathetic and parasympathetic nerve supplies to the parotid gland were lost. One patient (case 9) who did not undergo surgical ablation of the cervical sympathetic chain or the external carotid artery developed FBS. Two patients (cases 10 and 11) who underwent surgical ablation of the cervical sympathetic chain did not develop FBS. In the 1950s and 1960s, superior cervical ganglionectomy was performed by neurosurgeons as treatment for cerebrovascular disease. In 1955, Gardner and Abdullah [6] reported superior cervical ganglionectomy in detail. To assess differences between preganglionic and postganglionic sympathectomy in terms of their value in the treatment of certain vascular or atrophic lesions of the brain, the superior cervical sympathetic ganglion was excised on one side, while on the opposite side, only the preganglionic fibers
were excised, leaving the ganglion in situ. Incidentally, most of the patients had pain in the parotid region on eating beginning the second week after surgery. The report states: ‘‘The pain occurs with the first bite of food and after a matter of seconds disappears and usually does not return during the rest of the meal’’. This is so similar to FBS that we believe that they are describing FBS. A further important point is that this pain occurred only on the side on which the superior cervical ganglion had been excised, and never on the side of the preganglionic sympathectomy. The fact that the pain never occurs on the preganglionic sympathectomized side suggests that the decentralized superior cervical ganglion still has residual or autonomous activity; it is unclear whether the activity is residual or autonomous. This theory is consistent with two otherwise-inexplicable findings in the present study. In one patient with a sympathetic chain schwannoma (case 10), complete intracapsular enucleation was accomplished with preservation of a large part of the cervical sympathetic trunk. Although the patient developed Horner’s syndrome, the superior cervical ganglion was probably preserved. Another patient with a sympathetic chain schwannoma (case 11) underwent ligation of the cervical sympathetic chain 1 cm distal from the carotid bifurcation, and the superior cervical ganglion was probably preserved. For now, we cannot give clear explanation that the patients who underwent ablation of the sympathetic chain develop Horner’s syndrome without exception but not always develop FBS; however, our two patients (case10 and 11) and Gardner’s report strongly suggest that the residual or autonomous activity that the decentralized superior cervical ganglion has can prevent the patients from developing the FBS. There remains only one patient (case 9) whose FBS we are unable to explain. Cernea et al. [7] reported a case of FBS after proper resection of an enlarged styloid process. It is important for head and neck surgeons to keep in mind the
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
possibility of FBS as a complication after operations involving the PPS, and to operate carefully in that region.
5. Conclusions The present cases strongly support the theory that FBS is caused by loss of sympathetic innervation to the parotid gland from the superior cervical ganglion. In addition, they suggest that residual or autonomous activity of the superior cervical ganglion can prevent development of FBS even if the lower sympathetic trunk is ablated. There is no established therapy for FBS; consequently, it is particularly important to prevent the development of FBS. Surgeons who operate in the PPS should be aware of FBS, and should preserve the cervical sympathetic trunk and external carotid artery whenever possible. For patients with cervical sympathetic schwannoma, intracapsular enucleation may help to prevent the development of FBS.
113
References [1] Haubrich WS. The first-bite syndrome. Henry Ford Hosp Med J 1986;34:275–8. [2] Chiu AG, Cohen JI, Burningham AR, Andersen PE, Davidson BJ. First bite syndrome: a complication of surgery involving the parapharyngeal space. Head Neck 2002;24:996–9. [3] Netterville JL, Jackson CG, Miller FR, Wanamaker JR, Glasscock ME. Vagal paraganglioma: a review of 46 patients treated during a 20-year period. Arch Otolaryngol Head Neck Surg 1998;124: 1133–40. [4] Cohen SM, Burkey BB, Netterville JL. Surgical management of parapharyngeal space masses. Head Neck 2005;27:669–75. [5] Amonoo-Kuofi HS. Horner’s syndrome revisited: with an update of the central pathway. Clin Anat 1999;12:345–61. [6] Gardner WJ, Abdullah AF. Parotid pain following superior cervical ganglionectomy: a clinical example of the antagonistic action of the parasympathetic and sympathetic systems. Am J Med Sci 1955;230: 65–9. [7] Cernea CR, Hojaij FC, De Carlucci Jr D, Plopper C, Vanderley F, Guerreiro CA, et al. First-bite syndrome after resection of the styloid process. Laryngoscope 2007;117:181–2.
First-bite syndrome: A review of 29 patients with parapharyngeal space tumor Yoshiyuki Kawashima a,*, Takuro Sumi a, Taro Sugimoto a, Seiji Kishimoto b a
Department of Otolaryngology, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan b Department of Head and Neck Surgery, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8519, Japan Received 25 February 2007; accepted 25 June 2007 Available online 11 September 2007
Abstract Objective: The aim of this study is to clarify the etiology of first-bite syndrome. Methods: A total of 29 consecutive patients treated with a surgical resection for parapharyngeal space tumor between April 1999 and April 2005 were retrospectively reviewed. Results: Nine cases of first-bite syndrome were developed. Eight of those patients had undergone surgical ablation of the cervical sympathetic chain and/or external carotid artery. In two patients who underwent ablation of the sympathetic chain but in whom first-bite syndrome did not develop, the superior cervical ganglion was probably preserved. Conclusion: The present findings strongly indicate that loss of sympathetic innervation to the parotid gland from the superior cervical ganglion causes first-bite syndrome. They also suggest that some residual or autonomous activity of the superior cervical ganglion could prevent development of first-bite syndrome even if the lower sympathetic trunk was ablated. # 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: First-bite syndrome; Superior cervical ganglion; Parapharyngeal space; Complications
1. Introduction In 1986, Haubrich [1] first described first-bite syndrome (FBS). Typically, patients report excruciating pain in the parotid region only after the first bite of a meal. The pain improves with the subsequent masticatory movements of that meal, but invariably returns to the excruciating level at the first bite of the next meal [2]. The management of this syndrome can be troublesome. Treatment usually involves nonsteroidal anti-inflammatory medications or even carbamazepine, both of which provide poor results; all attempts at surgical treatment have been unsuccessful [2]. The pathogenesis of FBS is largely unknown, but researchers have proposed that it involves loss of sympathetic nerve innervation to the parotid gland after operations involving * Corresponding author. Tel.: +81 3 5803 5308; fax: +81 3 3813 2134. E-mail address: [email protected] (Y. Kawashima).
the parapharyngeal space (PPS), causing denervation supersensitivity of salivary gland myoepithelial cells [3]. We noticed that not all patients who underwent surgical ablation of the cervical sympathetic chain developed FBS, and hypothesized that patients who underwent surgical ablation of the cervical sympathetic chain but did not develop FBS could provide the key to understand the etiology of FBS. In the present study, we reviewed our patients who underwent PPS resection with or without subsequent occurrence of FBS, and reviewed the relevant literature.
2. Materials and methods We retrospectively reviewed the medical records of all patients who underwent surgery to remove a PPS tumor at Tokyo Medical and Dental University Hospital between
0385-8146/$ – see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.anl.2007.06.005
110
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
April 1999 and April 2005. The medical records were reviewed for the following information: patient demographics (age, sex); clinical manifestations; radiographic findings; methods of treatment, including surgical notes; histological reports and complications. To differentiate FBS from other sources of pain, we excluded from analysis all patients who had temporomandibular joint pain postoperatively or who were diagnosed with malignant tumors.
3. Results Twenty-nine patients with a PPS tumor were treated over a 7-year period (1999–2005). Of these 29 tumors, 6 (21%) were found to be malignant. Another patient had experienced severe trismus, temporomandibular joint pain, and dysphagia postoperatively. Thus, 7 of the 29 cases were excluded from the study, leaving 22 cases. The mean age of these 22 patients (10 men and 12 women) at the time of surgery was 40 years (range, 10–66 years). For each of the 22 patients, Table 1 shows whether FBS developed, its site of
origin, histology, size, surgical approach, ablated structures, and complications. Ten patients (45%) had pleomorphic adenomas arising from the deep lobe of the parotid gland or minor salivary gland; in 4 of these 10 patients, the adenoma was continuous with the deep lobe of the parotid gland. The remaining 12 cases were as follows: five patients (23%) had sympathetic chain schwannomas; two patients (9%) had vagus nerve schwannomas; one patient had an auriculotemporal nerve schwannoma; two patients (9%) had carotid body tumors (paragangliomas); one patient had an oncocytoma; and one patient had a lipoma. The mean major axis length of the tumors was 5.9 cm (range, 3–9 cm). Various surgical approaches were used to resect the tumors. A transcervical approach was used to resect 11 tumors: four sympathetic schwannomas, two vagus nerve schwannomas, two carotid body tumors, two pleomorphic adenomas, and one lipoma. A transcervical–transparotid approach was used to resect nine tumors: seven pleomorphic adenomas, one sympathetic nerve schwannoma, and one oncocytoma. A transcervical–transparotid approach with mandibulotomy was used to resect one auriculotemporal nerve schwannoma.
Table 1 Diagnosis, clinical features, and histological features
FBS, first-bite syndrome; C, transcervical; CP, transcervical–transparotid; CO, transcervical–transoral; M, mandibulotomy.
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
A transcervical–transoral approach was used to resect one pleomorphic adenoma. FBS developed in nine patients (41%; three men and six women), who had a median age of 42 years (range, 17–66 years). The mean major axis length of those nine tumors was 5.9 cm (range, 4–8 cm). A transcervical–transparotid approach was used to resect six of those tumors; a transcervical approach was used to resect the other three tumors. All nine patients had undergone surgical resection of structures within the PPS ipsilateral to the symptomatic side. None of the nine patients had a preoperative finding of FBS, Horner’s syndrome, or cranial nerve deficits. Three of the nine patients had sympathetic chain schwannoma. All three of those patients had undergone ablation of the cervical sympathetic chain and developed Horner’s syndrome postoperatively. The other six patients had pleomorphic adenoma arising from the deep lobe of the parotid gland or minor salivary gland; three of these six tumors were continuous with the deep lobe of the parotid gland. Patients with salivary gland tumors (cases 4–8) underwent ligation of the external carotid artery proximal to the deep surface of the parotid gland, and all five of those patients developed FBS. One patient (case 9) who underwent resection of the deep lobe of the parotid gland and did not undergo ligation of the external carotid artery also developed FBS. Two patients (cases 10 and 11) who underwent resection of a sympathetic chain schwannoma did not develop FBS. One patient (case 12) with an auriculotemporal nerve schwannoma underwent ligation of his external carotid artery, but did not develop FBS. Fig. 1 shows the relationships between development of FBS and ablated structures. The first thing we noticed is that in patients who underwent surgical ablation of the cervical sympathetic chain, FBS did not necessarily develop. With the exception of patient 12, FBS developed in patients who
Fig. 1. Relationships between development of first-bite syndrome and ablated structures. First-bite syndrome did not develop in all patients who underwent surgical ablation of the cervical sympathetic chain. With the exception of one patient (case 12), first-bite syndrome developed in patients who had undergone ligation of the external carotid artery.
111
underwent ligation of the external carotid artery. With the exception of patient 9, FBS did not develop in patients who did not undergo ablation of the sympathetic chain and/or external carotid artery.
4. Comment In 1986, Haubrich [1] first described FBS. Since 1998, when Netterville et al. [3] described FBS caused by PPS resection, FBS has been well known to head and neck surgeons. FBS is the most common of the various surgical complications of PPS surgery that have been reported in the literature [4]. The pain of FBS is due to severe cramping or spasm in the parotid gland region at the first bite of each meal; the pain diminishes over the next several bites [2]. The cause of FBS is largely unknown, but Netterville et al. [3] have proposed that FBS is caused by the loss of sympathetic innervation to the parotid gland. Before discussing the possible mechanisms of postoperative FBS, we should review the autonomous innervation of the parotid gland (Fig. 2). The parotid gland is innervated by both sympathetic and parasympathetic pathways. The sympathetic pathway can be regarded as a threeneuron pathway. The first-order neurons of the sympathetic pathway originate in the central nervous system (in the hypothalamus), and descend through the brainstem to a synapse in the thoracic region of the spinal cord. The second-order neurons exit the spinal cord at the first thoracic segment, and enter the sympathetic chain ascending through the inferior and middle cervical ganglia, synapsing in the superior cervical ganglion. The superior cervical ganglion is roughly 2.5 cm long and is located at the level of the second and third cervical vertebrae, posterior to the carotid sheath, between the internal carotid artery and the longus capitis muscle [5]. Anterior branches are distributed along the external and internal carotid arteries. Postganglionic nerves that innervate the eyeball, eyelid or orbit run in the internal carotid nerve and plexus. Most of the fibers that innervate the sweat glands or skin of the face or scalp run along the external carotid artery. The sympathetic nerve supply to the parotid gland itself branches from the external carotid artery plexus to travel along the middle meningeal artery. Thus, a loss of sympathetic innervation to the parotid gland can occur even in the absence of Horner’s syndrome. The parasympathetic innervation of the parotid gland consists mainly of ninth nerve impulses originating in the inferior salivary nucleus, which arrive at the otic ganglion via the glossopharyngeal nerve and tympanic plexus. From the otic ganglion, postganglionic fibers to the parotid gland pass to the neighboring auriculotemporal branch of the mandibular nerve. Netterville et al. [3] reviewed 46 vagal paragangliomas. In their series, nine patients exhibited FBS, and all nine of those patients had undergone resection of their sympathetic chain and/or exhibited loss of sympathetic function
112
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
Fig. 2. Pathway of autonomous nervous supply to the parotid gland.
manifested by Horner’s syndrome [3]. Two other patients underwent resection of the sympathetic trunk, and did not report parotid pain. Based on these findings, Netterville et al. [3] proposed that FBS was caused by damage or removal of the cervical sympathetic trunk with loss of sympathetic innervation to the parotid gland. In 2002, to assess the anatomical basis of this proposal, Chiu et al. [2] reviewed the records of 12 patients with FBS. Six patients exhibited postoperative Horner’s syndrome. The other six patients had undergone external carotid artery ligation inferior to the parotid gland. These two studies strongly suggest that FBS is caused by loss of sympathetic innervation of the parotid gland. Although most of the present cases strongly support the proposal of Netterville et al. [3], there are some exceptions (Fig. 1). One patient (case 12) who underwent ligation of the external carotid artery did not develop FBS. This patient, who had an auriculotemporal nerve schwannoma, underwent ligation of the auriculotemporal nerve as well as the external carotid artery. The reason why FBS did not develop in that patient is probably that both sympathetic and parasympathetic nerve supplies to the parotid gland were lost. One patient (case 9) who did not undergo surgical ablation of the cervical sympathetic chain or the external carotid artery developed FBS. Two patients (cases 10 and 11) who underwent surgical ablation of the cervical sympathetic chain did not develop FBS. In the 1950s and 1960s, superior cervical ganglionectomy was performed by neurosurgeons as treatment for cerebrovascular disease. In 1955, Gardner and Abdullah [6] reported superior cervical ganglionectomy in detail. To assess differences between preganglionic and postganglionic sympathectomy in terms of their value in the treatment of certain vascular or atrophic lesions of the brain, the superior cervical sympathetic ganglion was excised on one side, while on the opposite side, only the preganglionic fibers
were excised, leaving the ganglion in situ. Incidentally, most of the patients had pain in the parotid region on eating beginning the second week after surgery. The report states: ‘‘The pain occurs with the first bite of food and after a matter of seconds disappears and usually does not return during the rest of the meal’’. This is so similar to FBS that we believe that they are describing FBS. A further important point is that this pain occurred only on the side on which the superior cervical ganglion had been excised, and never on the side of the preganglionic sympathectomy. The fact that the pain never occurs on the preganglionic sympathectomized side suggests that the decentralized superior cervical ganglion still has residual or autonomous activity; it is unclear whether the activity is residual or autonomous. This theory is consistent with two otherwise-inexplicable findings in the present study. In one patient with a sympathetic chain schwannoma (case 10), complete intracapsular enucleation was accomplished with preservation of a large part of the cervical sympathetic trunk. Although the patient developed Horner’s syndrome, the superior cervical ganglion was probably preserved. Another patient with a sympathetic chain schwannoma (case 11) underwent ligation of the cervical sympathetic chain 1 cm distal from the carotid bifurcation, and the superior cervical ganglion was probably preserved. For now, we cannot give clear explanation that the patients who underwent ablation of the sympathetic chain develop Horner’s syndrome without exception but not always develop FBS; however, our two patients (case10 and 11) and Gardner’s report strongly suggest that the residual or autonomous activity that the decentralized superior cervical ganglion has can prevent the patients from developing the FBS. There remains only one patient (case 9) whose FBS we are unable to explain. Cernea et al. [7] reported a case of FBS after proper resection of an enlarged styloid process. It is important for head and neck surgeons to keep in mind the
Y. Kawashima et al. / Auris Nasus Larynx 35 (2008) 109–113
possibility of FBS as a complication after operations involving the PPS, and to operate carefully in that region.
5. Conclusions The present cases strongly support the theory that FBS is caused by loss of sympathetic innervation to the parotid gland from the superior cervical ganglion. In addition, they suggest that residual or autonomous activity of the superior cervical ganglion can prevent development of FBS even if the lower sympathetic trunk is ablated. There is no established therapy for FBS; consequently, it is particularly important to prevent the development of FBS. Surgeons who operate in the PPS should be aware of FBS, and should preserve the cervical sympathetic trunk and external carotid artery whenever possible. For patients with cervical sympathetic schwannoma, intracapsular enucleation may help to prevent the development of FBS.
113
References [1] Haubrich WS. The first-bite syndrome. Henry Ford Hosp Med J 1986;34:275–8. [2] Chiu AG, Cohen JI, Burningham AR, Andersen PE, Davidson BJ. First bite syndrome: a complication of surgery involving the parapharyngeal space. Head Neck 2002;24:996–9. [3] Netterville JL, Jackson CG, Miller FR, Wanamaker JR, Glasscock ME. Vagal paraganglioma: a review of 46 patients treated during a 20-year period. Arch Otolaryngol Head Neck Surg 1998;124: 1133–40. [4] Cohen SM, Burkey BB, Netterville JL. Surgical management of parapharyngeal space masses. Head Neck 2005;27:669–75. [5] Amonoo-Kuofi HS. Horner’s syndrome revisited: with an update of the central pathway. Clin Anat 1999;12:345–61. [6] Gardner WJ, Abdullah AF. Parotid pain following superior cervical ganglionectomy: a clinical example of the antagonistic action of the parasympathetic and sympathetic systems. Am J Med Sci 1955;230: 65–9. [7] Cernea CR, Hojaij FC, De Carlucci Jr D, Plopper C, Vanderley F, Guerreiro CA, et al. First-bite syndrome after resection of the styloid process. Laryngoscope 2007;117:181–2.