First Bite Syndrome After Bilateral Temporomandibular Joint Replacement: Case Report Nada Alwanni, DMD, MS,* Mehmet Ali Altay, DDS, PhD,y Dale A. Baur, DDS, MD,z and Faisal A. Quereshy, MD, DDSx First bite syndrome (FBS) refers to intense pain in the parotid region, which coincides with the first bite of every meal, gradually subsides over the next several bites, but returns with the first bite of the next meal. The definitive diagnosis can be readily established by the characteristic onset of pain after the first bite of every meal. Pain is typically most intense at the first meal of the day, although some patients experience symptoms when thinking of food or salivating. FBS is a recognized complication of surgery within the parapharyngeal space; however, other surgical procedures involving the upper neck have been associated with this syndrome. The extreme rarity of FBS complicates a thorough understanding of its pathophysiology. Various medical agents have been used, with variable success, for the management of patients with FBS. Although proved effective, more radical treatment modalities are commonly reserved for persistent or refractory cases, because there is potential of spontaneous decrease in the severity of symptoms with time. This report describes the case of a patient presenting with symptoms of FBS after bilateral temporomandibular joint replacement. To the authors’ knowledge, this is the first case of FBS in the literature occurring after temporomandibular joint replacement. Ó 2016 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 74:480-488, 2016
Initially described by Haubrich1 in 1986, first bite syndrome (FBS) refers to intense pain in the parotid region, which coincides with the first bite of every meal. The pain gradually subsides over the next several bites, but returns with the first bite of the next meal. The definitive diagnosis can be readily established by the characteristic onset of pain after the first bite of every meal. Pain is typically most intense at the first meal of the day, although some patients also experience symptoms when thinking of food or salivating. Substantial discomfort experienced
during meals understandably causes a worsened quality of life.2 FBS is a recognized complication of surgery within the parapharyngeal space; however, other surgical procedures involving the upper neck (resection of mixed or cervical sympathetic nerve tumors, deep cervical lymph node dissection, parotid gland surgery, infratemporal fossa surgery, internal carotid artery surgery, resection of the styloid process) have been associated with this syndrome.3 Despite the different pathogenic mechanisms that have been proposed, the extreme
*Research Fellow, Department of Oral and Maxillofacial Surgery,
Dr Baur is a paid consultant for Novartis Pharmaceuticals and
School of Dental Medicine, Case Western Reserve University,
Checkpoint Surgical LLC and Dr Altay provided consultancy for
Cleveland, OH. yAssistant Professor, Department of Oral and Maxillofacial
Checkpoint Surgical LLC in 2014. Address correspondence and reprint to Dr Baur: Department of
Surgery, Faculty of Dentistry, Akdeniz University, Antalya, Turkey.
Oral and Maxillofacial Surgery, School of Dental Medicine, Case
zAssociate Professor and Chair, Department of Oral and
Western Reserve University, 2124 Cornell Road, Cleveland, OH,
Maxillofacial Surgery, School of Dental Medicine, Case Western
44106-4905; e-mail: [email protected]
Reserve University, Cleveland, OH.
Received April 13 2015
xAssociate
Professor
and
Residency
Program
Director,
Accepted September 28 2015
Department of Oral and Maxillofacial Surgery, School of Dental
Ó 2016 American Association of Oral and Maxillofacial Surgeons
Medicine, Case Western Reserve University, Cleveland, OH.
0278-2391/15/01303-8 http://dx.doi.org/10.1016/j.joms.2015.09.034
480
481
ALWANNI ET AL
rarity of FBS complicates a thorough understanding of its pathophysiology, which remains to be elucidated. Various medical agents, including anti-inflammatory drugs, local or regional anesthetic blocks, oral analgesics, anticonvulsants, and tricyclic antidepressants, have been attempted, with variable success, for the management of patients with FBS. More recently, botulinum toxin (Botox) injection into the parotid gland has been used with promising results in a limited number of studies.4,5 Although previously proved to be effective, more radical treatment modalities reported in the literature, including neoadjuvant radiotherapy or neurectomy of the tympanic or auriculotemporal nerves, are commonly reserved for persistent or refractory cases, because there is potential of spontaneous decrease in the severity of symptoms with time.3,6-9 This report describes the case of a patient presenting with symptoms of FBS after bilateral temporomandibular joint (TMJ) replacement. To the authors’ knowledge, this is the first case of FBS in the literature occurring after TMJ replacement.
Report of Case A 34-year-old woman who previously underwent bilateral TMJ discectomy with a temporalis muscle flap after failed conservative treatment approaches, including dental splint therapy, medical therapy, and
arthrocentesis, presented to the Department of Oral and Maxillofacial Surgery, Case Western Reserve University School of Dental Medicine (Cleveland, OH) for the evaluation of bilateral TMJ pain, self-resolving closed lock, and limited mouth opening. The patient had an unremarkable medical history and was not taking any prescription medications. Her clinical examination showed a maximal incisal opening of 22 mm, severe limitation of lateral excursions of the mandible (right, 0 mm; left, 2 mm), and crepitus, with normal function of all cranial nerves. The radiologic evaluation of the patient was carried out with a computed tomogram and a previous magnetic resonance image, which identified degenerative changes of the TMJs, but no other concomitant pathology in the region. Several attempts for relief, including medical therapy and arthrocentesis, were performed without resolution of her symptoms. Taking the history of chronic pain and multiple interventions on the 2 joints without appreciable improvement and functional limitations into consideration, the decision was made to perform bilateral joint replacement. The patient received bilateral customalloplastic implants (TMJ Concepts, Ventura, CA) using submandibular Risdon and preauricular incisions and abdominal fat grafts (Figs 1, 2). The course of the surgery was uneventful, and the patient returned to function and was started on a soft diet. During a routine follow-up 2 weeks after the surgery, the patient complained of severe bilateral pain
FIGURE 1. Panoramic radiograph of the patient taken after bilateral temporomandibular joint replacement. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
482
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
FIGURE 2. Anteroposterior radiograph of the patient taken after bilateral temporomandibular joint replacement. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
across her lower jaw, which often radiated to the ear and neck, precipitated by eating and drinking. She described her pain as an electric shock–like nerve pain with the first bite of the food that subsided with each subsequent bite only to recur with the first bite of the next meal. She described the intensity of her pain as 10 on a scale of 0 to 10 (0, no pain at all; 10, the worst possible pain). The pain was reported to be of equal severity on the 2 sides. Clinical evaluation of the patient indicated normal function of the implants bilaterally and adequate range of motion. A stable occlusion with no posterior open bite and
appropriate healing with no signs of infection were observed. These findings were confirmed on subsequent follow-ups of the patient. Hardware loosening or failure was ruled out by clinically examining the occlusion, facial symmetry, and subjective symptoms and evaluating postoperative radiographs (Figs 1, 2). No tenderness was noted on lateral palpation of the prosthetic joints. All cranial nerve functions were found to be intact bilaterally, except for hypersensitivity when applying pressure over the incision lines, which was considered a normal finding during wound healing. No evidence of Frey
ALWANNI ET AL
syndrome was noted or was reported by the patient. In addition, there were no manifestations of Horner syndrome. With symptoms suggestive of FBS, all preoperative imaging studies were re-evaluated with particular interest in the parapharyngeal space and the parotid region. No evidence of pathology was noted because all examination findings were within normal limits. The diagnosis of FBS was strongly considered and gabapentin 300 mg 3 times daily was prescribed. During the first 3 months after surgery, the patient reported fear of experiencing the strong pain, which at times kept her from eating or drinking as desired. A gradual but limited improvement of symptoms was noted during the following few months with the gabapentin therapy. At the 7-month followup, the patient described the intensity of her ‘‘nerve pain’’ as 6 on a 10-point scale, although the classic symptoms of FBS persisted. She was able to better tolerate the symptoms of FBS and was generally able to eat a normal diet. At the time of this submission, the patient was 9 months past surgery, scored her pain as 3 on a 10-point scale, and reported that her quality of life has greatly improved with gabapentin therapy.
Discussion FBS is described as excruciating pain in the parotid region after the first bite of each meal that resolves with subsequent bites, only to recur with the first bite of the next meal.10 The pain, which can radiate to the ear, oral cavity, or the mandible, is often reported to be most intense during the first meal of the day. Symptoms are often mild, but can be severe enough to impair a patient’s ability to eat or drink and therefore negatively affect quality of life. A consensus has been reached in the literature on the fact that a confirmed diagnosis of FBS is exclusively clinical and based on findings of a clinical interview and normal physical examination findings.3 However, differential diagnoses to be considered include gastroesophageal reflux, glossopharyngeal neuralgia, and Eagle syndrome, which must be eliminated before initiating treatment. In the present case, these diagnoses were eliminated by physical and radiographic evaluations of the patient. Degenerative changes of the TMJ and TMJ dysfunction, which resolved immediately after the operation, also were excluded from the differential diagnosis. Although discussed in several studies, the pathophysiology of FBS remains the least understood aspect of this disease. Because of the particular interest in the present case, to clearly identify the origin, the authors reviewed the pathogenic mechanisms that were previously proposed for FBS in the literature. Initially postulated by Netterville et al,11 the common theory in the
483 literature involves the loss of sympathetic innervation to the ipsilateral parotid gland, leading to denervation of sympathetic receptors located on parotid myoepithelial cells. Parasympathetic receptors, which also exist on these cells, subsequently become hypersensitive, resulting in a very intense, contractile response at the first bite, inducing the excruciating pain described by patients.3,11 Subsequently, as supported by Chiu et al,6 this mechanism also was used to explain nonsurgical forms of FBS owing to malignant tumors of the parotid gland and the parapharyngeal space.12,13 In their retrospective review of 29 patients, Kawashima et al14 suggested that FBS results specifically from the loss of innervation from the superior cervical ganglion. Table 1 presents a summary of all articles that could be retrieved from Medline and PubMed using the key term first bite syndrome.2-38 It should be noted that 2 main etiologic factors for the development of FBS can be distinguished in the current literature3: surgery involving the upper neck and tumors of the parotid, parapharyngeal space, or submandibular gland. Among these, surgery involving the parapharyngeal space, particularly resection of the cervical sympathetic chain or ligation of sympathetic nerves along the vasculature toward the parotid gland, is more commonly associated with the development of FBS and currently is regarded as the main etiologic factor.4-8,14-27 Parotid gland surgery is another prevalent factor that has been linked with FBS by several studies.15,28-32 FBS also has been encountered as a finding of tumors of the submandibular gland,32 parotid gland,12 and parapharyngeal space,13,33,34 which draws attention to the importance of imaging assessments to rule out a malignancy when this syndrome occurs in the absence of a history of upper neck surgery. It is noteworthy that FBS has not been described previously as an outcome of alloplastic TMJ replacement surgery, which typically does not involve exposure of the parotid gland, parapharyngeal space, or cervical sympathetic chain. The present patient was free of clinical symptoms of cervical sympathetic disruption postoperatively, and radiographic evaluations failed to show impingement of the cervical sympathetic chain, carotid vasculature, or parotid by a mass. When these findings are considered, improvement of symptoms with gabapentin therapy lends support to a neurogenic pain mechanism. Another theory that can be made in this case is the compression injury of the postganglionic sympathetic fibers during retraction, which resulted in loss of sympathetic input to the parotid gland. The authors believe this etiologic mechanism is more conceivable because the Risdon approach with a preauricular incision was used without dissection of the parotid gland in this case. The gradual recovery of the patient
Year
Authors
Article Type
Number or Percentage of Patients With or Who Developed FBS
Surgery Performed and Occult Malignancy
2015
Casani et al28
Research
2 3
2015 2015
Scholey and Suida35 Mikolajczak et al29
Case report Case report
1 1
4
2015
Fiorini et al15
Case report
2
5
2014
Navaie et al16
Review
6
2014
Ansarin et al17
Case report
7
2014
Houle and Mandel30
Case report
8
2014
Horowitz et al18
Research
9
2014
Amin et al31
Case report
1
Transoral robotic surgical removal of retrostyloid PPS schwannoma Parotid gland deep lobectomy Excision of PPS pleomorphic adenomas by transparotid transcervical approach Left total parotidectomy
10 11 12
2013 2013 2013
Laccourreye et al3 Abdeldaoui et al19 Wang et al38
Review Research Case report
Review 17 1
Review Upper cervical surgery Carotid endarterectomy
3 of 24
21.1% of postsurgical adverse events were FBS alone and 15.7% were FBS and Horner syndrome 1 of 2 patients developed FBS 3 mo after surgery 1 1 of 13
Surgery for benign tumors of parotid gland Bimaxillary osteotomy Partial parotidectomy
Patient 1, total parotidectomy with facial nerve preservation; patient 2, extracapsular dissection of PPS neurinoma Resection of cervical sympathetic chain schwannomas
Success Rate and Outcomes Spontaneous resolution
None Intraparotid injections of botulinum toxin type A 6 sessions of 30 minutes of acupuncture/wk
Self-resolved after 3 mo complete resolution
Patient 1, improvement; patient 2, improvement
Botulinum toxin therapy refused by patient
Laser tympanic plexus ablation Review Varied Botulinum toxin A parotid injection, 50 U total
Complete resolution 2 mo after surgery Review Varied Partial improvement 1 mo after injection
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
1
Treatment Rendered
484
Table 1. SUMMARY OF ALL ARTICLES THAT WERE RETRIEVED FROM THE MEDLINE AND PUBMED DATABASE USING THE KEY TERM FIRST BITE SYNDROME
2013
Guss et al32
Case report
1
Adenoid cystic carcinoma of submandibular gland
14
2013
Sims and Suen5
Case report
3
15
2012
Linkov et al20
Research
16
2011
Costa et al8
Case report
2
17
2011
Wong et al9
Case report
1
Patient 1, tongue and upper neck surgery; patient 2, PPS surgery; patient 3, right tongue and external carotid surgery Surgery of deep lobe of parotid gland, PPS, or ITF Patient 1, extended radical neck dissection and PPS; patient 2, ligation of external carotid artery Carotid endarterectomy
18
2011
Diercks et al12
Case report
1
19
2011
Deganello et al13
Case report
1
20
2011
Lieberman and Har-El34
Case report
1
PPS monophasic synovial sarcoma
21
2011
Albasri et al10
Case report
2
Patient 1, right carotid endarterectomy; patient 2, resection of left-side cervical paraganglioma
22 23 24
2009 2009 2009
Chijawa et al21 Borras-Perera et al33 Lee et al37
Research Case report Research
7 of 22 1 5
PPS tumor surgery Parapharyngeal tumor Head and neck surgery
25
2009
Casserly et al22
Case report
1
45 of 499
Mucoepidermoid carcinoma of parotid gland Parapharyngeal adenoid cystic carcinoma
Pain persisted after surgery but resolved after RT
Varied
Partial resolution in 69%, complete resolution in 12% of patients Partial improvement with medical therapy, complete resolution after RT
NSAID and acetaminophen followed by adjuvant RT as part of cancer treatment Patient refused treatment Excision of tumor
Complete resolution in 2 cases and near complete resolution in third case
Pain gradually resolved over 6 mo Resolution
Total parotidectomy, upper neck surgery and adjuvant RT Resection of tumor and parotid gland and sympathetic chain Patient 1, amitriptyline; patient 2, carbamazepine and gabapentin, regional local anesthetic blocks with bupivacaine
Resolution
Carbamazepine Intraparotid injection of botulinum toxin type A Pregabalin
No improvement Considerable improvement in 1-3 mo Resolution
Resolution
Patient 1, limited relief followed by complete resolution at 12 mo; patient 2, limited relief followed by complete resolution at 18 mo
485
Surgery of cervical sympathetic chain schwannoma
Excision of submandibular gland and nearby lymph nodes, followed by RT Botulinum toxin injection into parotid gland; units and intervals varied
ALWANNI ET AL
13
486
Table 1. Cont’d
Year
Authors
Article Type
Number or Percentage of Patients With or Who Developed FBS
2009
Phillips and FarquharSmith23
Case report
1
27
2008
Ali et al4
Case report
1
28
2008
Mandel and Syrop24
Case report
1
29
2007
Lin et al25
Case report
1 of 3
30
2008
Kawashima et al14
Research
9 of 29
31
2007
Kamal et al7
Case report
1
32
2007
Cernea et al2
Case report
1
33
2006
Smith et al27
Research
34 35
2005 2004
Sharma and Massey36 Wax et al26
Review Case series
36
2002
Chiu et al6
Research
37
1998
Netterville et al11
Case series
7.27% of complications after CRB and 12.5% after CBR-VASC Review 2 of 4
12
9 of 40
Treatment Rendered
Success Rate and Outcomes
Resection of benign paraganglioma of parapharyngeal area Resection of right neck lymphangioma PPS surgery for cervical schwannoma Surgical excision of cervical sympathetic chain schwannoma Surgical resection of PPS tumor Surgical excision of cervical sympathetic chain schwannoma Resection of styloid process CBR
Carbamazepine and amitriptyline
Partial resolution
Botulinum toxin
Complete resolution within 10 wk
Carbamazepine
Resolution in 2 yr
Review Surgical excision of cervical sympathetic chain schwannoma PPS surgery
Review
Review Partial resolution over time
Radiation, NSAIDs, carbamazepine, tympanic neurectomy varied
Varied
Vagal paraganglioma treated surgically
Abbreviations: CBR, carotid body tumor resection; CBR-VASC, carotid body tumor resection requiring vascular reconstruction; FBS, first bite syndrome; ITF, infratemporal fossa; NSAID, nonsteroidal anti-inflammatory drug; PPS, parapharyngeal space; RT, radiotherapy. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
26
Surgery Performed and Occult Malignancy
487
ALWANNI ET AL
similarly supports the theory that the FBS originated from a reversible neural injury. In a large number of FBS cases, the severity of pain decreases spontaneously and often completely disappears in several months to 1 year.3,6,19,35 However, in certain cases, the pain can substantially affect quality of life and physical health by preventing the patient from eating or drinking, which results in inadequate nutritional intake. The present patient reported severe pain triggered by the first bite or sip of food and certain drinks in the lower third of the face that prevented her from eating and drinking, with subsequent weight loss. Although spontaneous resolution of symptoms has been reported, the authors believe that severe impairment in quality of life necessitates immediate treatment when FBS is encountered. Several medical2,6,8,10,22,23,33,36 and 6,12,13,31,32,34 surgical methods have been proposed for the management of FBS. Among these, anticonvulsants (gabapentin, pregabalin, carbamazepine) alone2,6,22,33,36 or in combination with tricyclic antidepressants (amitriptyline)10,23 have been reported to decrease the intensity of pain in a limited number of studies.16,17 Surgical management of FBS also has been attempted by some groups.6,12,13,31,32,34 Netterville et al11 reported excellent results in 1 patient after removal of the auriculotemporal nerve, whereas Lieberman and Har-El34 reported complete resolution of symptoms after total parotidectomy during treatment of a patient with parapharyngeal space malignancy. In addition, neoadjuvant radiotherapy has been reported to aid in resolution of postoperative FBS after neck lymph node dissection by several researchers.6,13,32 In their retrospective cohort study, which reviewed records of 499 patients who underwent surgery of the parapharyngeal space, deep lobe of the parotid gland, and the infratemporal fossa, Linkov et al20 identified an inverse relation between a history of radiation therapy and the development of FBS. However, the authors believe that such a radical approach should be reserved for cases in which radiotherapy is indicated owing to a malignancy, independent of FBS, or for cases in which all other conservative measures have failed. Another approach that has been reported for the management of FBS is intraparotid injection of Botox,4,5,20,29,37,38 although a standard regimen has not been defined.3,4,20 The authors’ experience with this case indicates the need for a treatment approach that should be tailored individually when managing patients with FBS. The present patient was managed conservatively, with improvement of symptoms with medical therapy alone, which the authors recommend as first-line treatment in this subset of patients.
A rare and therefore under-recognized complication of surgery involving the upper neck, FBS also can occur as a complication of alloplastic TMJ replacement. The definitive pathogenesis remains obscure, which supports the theory that the sympathetic and parasympathetic disruption on the myoepithelial cells of the parotid might not be the only contributing factor of FBS. Surgeons performing TMJ replacement procedures should be aware of this potential complication and its outcomes.
References 1. Haubrich WS: The first-bite syndrome. Henry Ford Hosp Med J 34:275, 1986 2. Cernea CR, Hojaij FC, De Carlucci D Jr, et al: First-bite syndrome after resection of the styloid process. Laryngoscope 117:181, 2007 3. Laccourreye O, Werner A, Garcia D, et al: First bite syndrome. Eur Ann Otorhinolaryngol Head Neck Dis 130:269, 2013 4. Ali MJ, Orloff LA, Lustig LR, et al: Botulinum toxin in the treatment of first bite syndrome. Otolaryngol Head Neck Surg 139: 742, 2008 5. Sims JR, Suen JY: First bite syndrome: Case report of three patients treated with botulinum toxin and review of other treatment modalities. Head Neck 35:E288, 2013 6. Chiu AG, Cohen JI, Burningham AR, et al: First bite syndrome: A complication of surgery involving the parapharyngeal space. Head Neck 24:996, 2002 7. Kamal A, Abd El-Fattah AM, Tawfik A, et al: Cervical sympathetic schwannoma with postoperative first bite syndrome. Eur Arch Otorhinolaryngol 264:1109, 2007 8. Costa TP, de Araujo CE, Filipe J, et al: First-bite syndrome in oncologic patients. Eur Arch Otorhinolaryngol 268:1241, 2011 9. Wong EH, Farrier JN, Cooper DG: First-bite syndrome complicating carotid endarterectomy: A case report and literature review. Vasc Endovascular Surg 45:459, 2011 10. Albasri H, Eley KA, Saeed NR: Chronic pain related to first bite syndrome: Report of two cases. Br J Oral Maxillofac Surg 49: 154, 2011 11. Netterville JL, Jackson CG, Miller FR, et al: Vagal paraganglioma: A review of 46 patients treated during a 20-year period. Arch Otolaryngol Head Neck Surg 124:1133, 1998 12. Diercks GR, Rosow DE, Prasad M, et al: A case of preoperative ‘ first-bite syndrome’’ associated with mucoepidermoid carcinoma of the parotid gland. Laryngoscope 121:760, 2011 13. Deganello A, Meccariello G, Busoni M, et al: First bite syndrome as presenting symptom of parapharyngeal adenoid cystic carcinoma. J Laryngol Otol 125:428, 2011 14. Kawashima Y, Sumi T, Sugimoto T, et al: First-bite syndrome: A review of 29 patients with parapharyngeal space tumor. Auris Nasus Larynx 35:109, 2008 15. Fiorini FR, Santoro R, Cristofaro G, et al: Potential use of acupuncture in the treatment of first bite syndrome. Am J Otolaryngol 36:484, 2015 16. Navaie M, Sharghi LH, Cho-Reyes S, et al: Diagnostic approach, treatment, and outcomes of cervical sympathetic chain schwannomas: A global narrative review. Otolaryngol Head Neck Surg 151:899, 2014 17. Ansarin M, Tagliabue M, Chu F, et al: Transoral robotic surgery in retrostyloid parapharyngeal space schwannomas. Case Rep Otolaryngol 2014:296025, 2014 18. Horowitz G, Ben-Ari O, Wasserzug O, et al: The transcervical approach for parapharyngeal space pleomorphic adenomas: Indications and technique. PLoS One 9:e90210, 2014 19. Abdeldaoui A, Oker N, Duet M, et al: First bite syndrome: A little known complication of upper cervical surgery. Eur Ann Otorhinolaryngol Head Neck Dis 130:123, 2013 20. Linkov G, Morris LG, Shah JP, et al: First bite syndrome: Incidence, risk factors, treatment, and outcomes. Laryngoscope 122:1773, 2012
488 21. Chijiwa H, Mihoki T, Shin B, et al: Clinical study of parapharyngeal space tumours. J Laryngol Otol Suppl 31:100, 2009 22. Casserly P, Kiely P, Fenton JE: Cervical sympathetic chain schwannoma masquerading as a carotid body tumour with a postoperative complication of first-bite syndrome. Eur Arch Otorhinolaryngol 266:1659, 2009 23. Phillips TJ, Farquhar-Smith WP: Pharmacological treatment of a patient with first-bite syndrome. Anaesthesia 64:97, 2009 24. Mandel L, Syrop SB: First-bite syndrome after parapharyngeal surgery for cervical schwannoma. J Am Dent Assoc 139:1480, 2008 25. Lin CC, Wang CC, Liu SA, et al: Cervical sympathetic chain schwannoma. J Formos Med Assoc 106:956, 2007 26. Wax MK, Shiley SG, Robinson JL, et al: Cervical sympathetic chain schwannoma. Laryngoscope 114:2210, 2004 27. Smith JJ, Passman MA, Dattilo JB, et al: Carotid body tumor resection: Does the need for vascular reconstruction worsen outcome? Ann Vasc Surg 20:435, 2006 28. Casani AP, Cerchiai N, Dallan I, et al: Benign tumours affecting the deep lobe of the parotid gland: How to select the optimal surgical approach. Acta Otorhinolaryngol Ital 35:80, 2015 29. Mikolajczak S, Ludwig L, Grosheva M, et al: First bite syndrome: Successful treatment with Botulinum toxin A. Laryngorhinootologie 94:924, 2015 (in German)
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT 30. Houle A, Mandel L: First bite syndrome after deep lobe parotidectomy: Case report. J Oral Maxillofac Surg 72:1475, 2014 31. Amin N, Pelser A, Weighill J: First bite syndrome: Our experience of laser tympanic plexus ablation. J Laryngol Otol 128: 166, 2014 32. Guss J, Ashton-Sager AL, Fong BP: First bite syndrome caused by adenoid cystic carcinoma of the submandibular gland. Laryngoscope 123:426, 2013 33. Borras-Perera M, Fortuny-Llanses JC, Palomar-Asenjo V, et al: First-bite syndrome. Acta Otorrinolaringol Esp 60:144, 2009 34. Lieberman SM, Har-El G: First bite syndrome as a presenting symptom of a parapharyngeal space malignancy. Head Neck 33:1539, 2011 35. Scholey AL, Suida MI: First bite syndrome after bimaxillary osteotomy: Case report. Br J Oral Maxillofac Surg 53:561, 2015 36. Sharma PK, Massey BL: Gabapentin for the treatment of first bite syndrome following parapharyngeal space surgery. J Otolaryngol Head Neck Surg 133:P173, 2005 37. Lee BJ, Lee JC, Lee YO, et al: Novel treatment of first bite syndrome using botulinum toxin type A. Head Neck 31:989, 2009 38. Wang TK, Bhamidipaty V, MacCormick M: First bite syndrome following ipsilateral carotid endarterectomy. Vasc Endovascular Surg 47:148, 2013
Initially described by Haubrich1 in 1986, first bite syndrome (FBS) refers to intense pain in the parotid region, which coincides with the first bite of every meal. The pain gradually subsides over the next several bites, but returns with the first bite of the next meal. The definitive diagnosis can be readily established by the characteristic onset of pain after the first bite of every meal. Pain is typically most intense at the first meal of the day, although some patients also experience symptoms when thinking of food or salivating. Substantial discomfort experienced
during meals understandably causes a worsened quality of life.2 FBS is a recognized complication of surgery within the parapharyngeal space; however, other surgical procedures involving the upper neck (resection of mixed or cervical sympathetic nerve tumors, deep cervical lymph node dissection, parotid gland surgery, infratemporal fossa surgery, internal carotid artery surgery, resection of the styloid process) have been associated with this syndrome.3 Despite the different pathogenic mechanisms that have been proposed, the extreme
*Research Fellow, Department of Oral and Maxillofacial Surgery,
Dr Baur is a paid consultant for Novartis Pharmaceuticals and
School of Dental Medicine, Case Western Reserve University,
Checkpoint Surgical LLC and Dr Altay provided consultancy for
Cleveland, OH. yAssistant Professor, Department of Oral and Maxillofacial
Checkpoint Surgical LLC in 2014. Address correspondence and reprint to Dr Baur: Department of
Surgery, Faculty of Dentistry, Akdeniz University, Antalya, Turkey.
Oral and Maxillofacial Surgery, School of Dental Medicine, Case
zAssociate Professor and Chair, Department of Oral and
Western Reserve University, 2124 Cornell Road, Cleveland, OH,
Maxillofacial Surgery, School of Dental Medicine, Case Western
44106-4905; e-mail: [email protected]
Reserve University, Cleveland, OH.
Received April 13 2015
xAssociate
Professor
and
Residency
Program
Director,
Accepted September 28 2015
Department of Oral and Maxillofacial Surgery, School of Dental
Ó 2016 American Association of Oral and Maxillofacial Surgeons
Medicine, Case Western Reserve University, Cleveland, OH.
0278-2391/15/01303-8 http://dx.doi.org/10.1016/j.joms.2015.09.034
480
481
ALWANNI ET AL
rarity of FBS complicates a thorough understanding of its pathophysiology, which remains to be elucidated. Various medical agents, including anti-inflammatory drugs, local or regional anesthetic blocks, oral analgesics, anticonvulsants, and tricyclic antidepressants, have been attempted, with variable success, for the management of patients with FBS. More recently, botulinum toxin (Botox) injection into the parotid gland has been used with promising results in a limited number of studies.4,5 Although previously proved to be effective, more radical treatment modalities reported in the literature, including neoadjuvant radiotherapy or neurectomy of the tympanic or auriculotemporal nerves, are commonly reserved for persistent or refractory cases, because there is potential of spontaneous decrease in the severity of symptoms with time.3,6-9 This report describes the case of a patient presenting with symptoms of FBS after bilateral temporomandibular joint (TMJ) replacement. To the authors’ knowledge, this is the first case of FBS in the literature occurring after TMJ replacement.
Report of Case A 34-year-old woman who previously underwent bilateral TMJ discectomy with a temporalis muscle flap after failed conservative treatment approaches, including dental splint therapy, medical therapy, and
arthrocentesis, presented to the Department of Oral and Maxillofacial Surgery, Case Western Reserve University School of Dental Medicine (Cleveland, OH) for the evaluation of bilateral TMJ pain, self-resolving closed lock, and limited mouth opening. The patient had an unremarkable medical history and was not taking any prescription medications. Her clinical examination showed a maximal incisal opening of 22 mm, severe limitation of lateral excursions of the mandible (right, 0 mm; left, 2 mm), and crepitus, with normal function of all cranial nerves. The radiologic evaluation of the patient was carried out with a computed tomogram and a previous magnetic resonance image, which identified degenerative changes of the TMJs, but no other concomitant pathology in the region. Several attempts for relief, including medical therapy and arthrocentesis, were performed without resolution of her symptoms. Taking the history of chronic pain and multiple interventions on the 2 joints without appreciable improvement and functional limitations into consideration, the decision was made to perform bilateral joint replacement. The patient received bilateral customalloplastic implants (TMJ Concepts, Ventura, CA) using submandibular Risdon and preauricular incisions and abdominal fat grafts (Figs 1, 2). The course of the surgery was uneventful, and the patient returned to function and was started on a soft diet. During a routine follow-up 2 weeks after the surgery, the patient complained of severe bilateral pain
FIGURE 1. Panoramic radiograph of the patient taken after bilateral temporomandibular joint replacement. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
482
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
FIGURE 2. Anteroposterior radiograph of the patient taken after bilateral temporomandibular joint replacement. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
across her lower jaw, which often radiated to the ear and neck, precipitated by eating and drinking. She described her pain as an electric shock–like nerve pain with the first bite of the food that subsided with each subsequent bite only to recur with the first bite of the next meal. She described the intensity of her pain as 10 on a scale of 0 to 10 (0, no pain at all; 10, the worst possible pain). The pain was reported to be of equal severity on the 2 sides. Clinical evaluation of the patient indicated normal function of the implants bilaterally and adequate range of motion. A stable occlusion with no posterior open bite and
appropriate healing with no signs of infection were observed. These findings were confirmed on subsequent follow-ups of the patient. Hardware loosening or failure was ruled out by clinically examining the occlusion, facial symmetry, and subjective symptoms and evaluating postoperative radiographs (Figs 1, 2). No tenderness was noted on lateral palpation of the prosthetic joints. All cranial nerve functions were found to be intact bilaterally, except for hypersensitivity when applying pressure over the incision lines, which was considered a normal finding during wound healing. No evidence of Frey
ALWANNI ET AL
syndrome was noted or was reported by the patient. In addition, there were no manifestations of Horner syndrome. With symptoms suggestive of FBS, all preoperative imaging studies were re-evaluated with particular interest in the parapharyngeal space and the parotid region. No evidence of pathology was noted because all examination findings were within normal limits. The diagnosis of FBS was strongly considered and gabapentin 300 mg 3 times daily was prescribed. During the first 3 months after surgery, the patient reported fear of experiencing the strong pain, which at times kept her from eating or drinking as desired. A gradual but limited improvement of symptoms was noted during the following few months with the gabapentin therapy. At the 7-month followup, the patient described the intensity of her ‘‘nerve pain’’ as 6 on a 10-point scale, although the classic symptoms of FBS persisted. She was able to better tolerate the symptoms of FBS and was generally able to eat a normal diet. At the time of this submission, the patient was 9 months past surgery, scored her pain as 3 on a 10-point scale, and reported that her quality of life has greatly improved with gabapentin therapy.
Discussion FBS is described as excruciating pain in the parotid region after the first bite of each meal that resolves with subsequent bites, only to recur with the first bite of the next meal.10 The pain, which can radiate to the ear, oral cavity, or the mandible, is often reported to be most intense during the first meal of the day. Symptoms are often mild, but can be severe enough to impair a patient’s ability to eat or drink and therefore negatively affect quality of life. A consensus has been reached in the literature on the fact that a confirmed diagnosis of FBS is exclusively clinical and based on findings of a clinical interview and normal physical examination findings.3 However, differential diagnoses to be considered include gastroesophageal reflux, glossopharyngeal neuralgia, and Eagle syndrome, which must be eliminated before initiating treatment. In the present case, these diagnoses were eliminated by physical and radiographic evaluations of the patient. Degenerative changes of the TMJ and TMJ dysfunction, which resolved immediately after the operation, also were excluded from the differential diagnosis. Although discussed in several studies, the pathophysiology of FBS remains the least understood aspect of this disease. Because of the particular interest in the present case, to clearly identify the origin, the authors reviewed the pathogenic mechanisms that were previously proposed for FBS in the literature. Initially postulated by Netterville et al,11 the common theory in the
483 literature involves the loss of sympathetic innervation to the ipsilateral parotid gland, leading to denervation of sympathetic receptors located on parotid myoepithelial cells. Parasympathetic receptors, which also exist on these cells, subsequently become hypersensitive, resulting in a very intense, contractile response at the first bite, inducing the excruciating pain described by patients.3,11 Subsequently, as supported by Chiu et al,6 this mechanism also was used to explain nonsurgical forms of FBS owing to malignant tumors of the parotid gland and the parapharyngeal space.12,13 In their retrospective review of 29 patients, Kawashima et al14 suggested that FBS results specifically from the loss of innervation from the superior cervical ganglion. Table 1 presents a summary of all articles that could be retrieved from Medline and PubMed using the key term first bite syndrome.2-38 It should be noted that 2 main etiologic factors for the development of FBS can be distinguished in the current literature3: surgery involving the upper neck and tumors of the parotid, parapharyngeal space, or submandibular gland. Among these, surgery involving the parapharyngeal space, particularly resection of the cervical sympathetic chain or ligation of sympathetic nerves along the vasculature toward the parotid gland, is more commonly associated with the development of FBS and currently is regarded as the main etiologic factor.4-8,14-27 Parotid gland surgery is another prevalent factor that has been linked with FBS by several studies.15,28-32 FBS also has been encountered as a finding of tumors of the submandibular gland,32 parotid gland,12 and parapharyngeal space,13,33,34 which draws attention to the importance of imaging assessments to rule out a malignancy when this syndrome occurs in the absence of a history of upper neck surgery. It is noteworthy that FBS has not been described previously as an outcome of alloplastic TMJ replacement surgery, which typically does not involve exposure of the parotid gland, parapharyngeal space, or cervical sympathetic chain. The present patient was free of clinical symptoms of cervical sympathetic disruption postoperatively, and radiographic evaluations failed to show impingement of the cervical sympathetic chain, carotid vasculature, or parotid by a mass. When these findings are considered, improvement of symptoms with gabapentin therapy lends support to a neurogenic pain mechanism. Another theory that can be made in this case is the compression injury of the postganglionic sympathetic fibers during retraction, which resulted in loss of sympathetic input to the parotid gland. The authors believe this etiologic mechanism is more conceivable because the Risdon approach with a preauricular incision was used without dissection of the parotid gland in this case. The gradual recovery of the patient
Year
Authors
Article Type
Number or Percentage of Patients With or Who Developed FBS
Surgery Performed and Occult Malignancy
2015
Casani et al28
Research
2 3
2015 2015
Scholey and Suida35 Mikolajczak et al29
Case report Case report
1 1
4
2015
Fiorini et al15
Case report
2
5
2014
Navaie et al16
Review
6
2014
Ansarin et al17
Case report
7
2014
Houle and Mandel30
Case report
8
2014
Horowitz et al18
Research
9
2014
Amin et al31
Case report
1
Transoral robotic surgical removal of retrostyloid PPS schwannoma Parotid gland deep lobectomy Excision of PPS pleomorphic adenomas by transparotid transcervical approach Left total parotidectomy
10 11 12
2013 2013 2013
Laccourreye et al3 Abdeldaoui et al19 Wang et al38
Review Research Case report
Review 17 1
Review Upper cervical surgery Carotid endarterectomy
3 of 24
21.1% of postsurgical adverse events were FBS alone and 15.7% were FBS and Horner syndrome 1 of 2 patients developed FBS 3 mo after surgery 1 1 of 13
Surgery for benign tumors of parotid gland Bimaxillary osteotomy Partial parotidectomy
Patient 1, total parotidectomy with facial nerve preservation; patient 2, extracapsular dissection of PPS neurinoma Resection of cervical sympathetic chain schwannomas
Success Rate and Outcomes Spontaneous resolution
None Intraparotid injections of botulinum toxin type A 6 sessions of 30 minutes of acupuncture/wk
Self-resolved after 3 mo complete resolution
Patient 1, improvement; patient 2, improvement
Botulinum toxin therapy refused by patient
Laser tympanic plexus ablation Review Varied Botulinum toxin A parotid injection, 50 U total
Complete resolution 2 mo after surgery Review Varied Partial improvement 1 mo after injection
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
1
Treatment Rendered
484
Table 1. SUMMARY OF ALL ARTICLES THAT WERE RETRIEVED FROM THE MEDLINE AND PUBMED DATABASE USING THE KEY TERM FIRST BITE SYNDROME
2013
Guss et al32
Case report
1
Adenoid cystic carcinoma of submandibular gland
14
2013
Sims and Suen5
Case report
3
15
2012
Linkov et al20
Research
16
2011
Costa et al8
Case report
2
17
2011
Wong et al9
Case report
1
Patient 1, tongue and upper neck surgery; patient 2, PPS surgery; patient 3, right tongue and external carotid surgery Surgery of deep lobe of parotid gland, PPS, or ITF Patient 1, extended radical neck dissection and PPS; patient 2, ligation of external carotid artery Carotid endarterectomy
18
2011
Diercks et al12
Case report
1
19
2011
Deganello et al13
Case report
1
20
2011
Lieberman and Har-El34
Case report
1
PPS monophasic synovial sarcoma
21
2011
Albasri et al10
Case report
2
Patient 1, right carotid endarterectomy; patient 2, resection of left-side cervical paraganglioma
22 23 24
2009 2009 2009
Chijawa et al21 Borras-Perera et al33 Lee et al37
Research Case report Research
7 of 22 1 5
PPS tumor surgery Parapharyngeal tumor Head and neck surgery
25
2009
Casserly et al22
Case report
1
45 of 499
Mucoepidermoid carcinoma of parotid gland Parapharyngeal adenoid cystic carcinoma
Pain persisted after surgery but resolved after RT
Varied
Partial resolution in 69%, complete resolution in 12% of patients Partial improvement with medical therapy, complete resolution after RT
NSAID and acetaminophen followed by adjuvant RT as part of cancer treatment Patient refused treatment Excision of tumor
Complete resolution in 2 cases and near complete resolution in third case
Pain gradually resolved over 6 mo Resolution
Total parotidectomy, upper neck surgery and adjuvant RT Resection of tumor and parotid gland and sympathetic chain Patient 1, amitriptyline; patient 2, carbamazepine and gabapentin, regional local anesthetic blocks with bupivacaine
Resolution
Carbamazepine Intraparotid injection of botulinum toxin type A Pregabalin
No improvement Considerable improvement in 1-3 mo Resolution
Resolution
Patient 1, limited relief followed by complete resolution at 12 mo; patient 2, limited relief followed by complete resolution at 18 mo
485
Surgery of cervical sympathetic chain schwannoma
Excision of submandibular gland and nearby lymph nodes, followed by RT Botulinum toxin injection into parotid gland; units and intervals varied
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13
486
Table 1. Cont’d
Year
Authors
Article Type
Number or Percentage of Patients With or Who Developed FBS
2009
Phillips and FarquharSmith23
Case report
1
27
2008
Ali et al4
Case report
1
28
2008
Mandel and Syrop24
Case report
1
29
2007
Lin et al25
Case report
1 of 3
30
2008
Kawashima et al14
Research
9 of 29
31
2007
Kamal et al7
Case report
1
32
2007
Cernea et al2
Case report
1
33
2006
Smith et al27
Research
34 35
2005 2004
Sharma and Massey36 Wax et al26
Review Case series
36
2002
Chiu et al6
Research
37
1998
Netterville et al11
Case series
7.27% of complications after CRB and 12.5% after CBR-VASC Review 2 of 4
12
9 of 40
Treatment Rendered
Success Rate and Outcomes
Resection of benign paraganglioma of parapharyngeal area Resection of right neck lymphangioma PPS surgery for cervical schwannoma Surgical excision of cervical sympathetic chain schwannoma Surgical resection of PPS tumor Surgical excision of cervical sympathetic chain schwannoma Resection of styloid process CBR
Carbamazepine and amitriptyline
Partial resolution
Botulinum toxin
Complete resolution within 10 wk
Carbamazepine
Resolution in 2 yr
Review Surgical excision of cervical sympathetic chain schwannoma PPS surgery
Review
Review Partial resolution over time
Radiation, NSAIDs, carbamazepine, tympanic neurectomy varied
Varied
Vagal paraganglioma treated surgically
Abbreviations: CBR, carotid body tumor resection; CBR-VASC, carotid body tumor resection requiring vascular reconstruction; FBS, first bite syndrome; ITF, infratemporal fossa; NSAID, nonsteroidal anti-inflammatory drug; PPS, parapharyngeal space; RT, radiotherapy. Alwanni et al. First Bite Syndrome After TMJ Replacement. J Oral Maxillofac Surg 2016.
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT
26
Surgery Performed and Occult Malignancy
487
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similarly supports the theory that the FBS originated from a reversible neural injury. In a large number of FBS cases, the severity of pain decreases spontaneously and often completely disappears in several months to 1 year.3,6,19,35 However, in certain cases, the pain can substantially affect quality of life and physical health by preventing the patient from eating or drinking, which results in inadequate nutritional intake. The present patient reported severe pain triggered by the first bite or sip of food and certain drinks in the lower third of the face that prevented her from eating and drinking, with subsequent weight loss. Although spontaneous resolution of symptoms has been reported, the authors believe that severe impairment in quality of life necessitates immediate treatment when FBS is encountered. Several medical2,6,8,10,22,23,33,36 and 6,12,13,31,32,34 surgical methods have been proposed for the management of FBS. Among these, anticonvulsants (gabapentin, pregabalin, carbamazepine) alone2,6,22,33,36 or in combination with tricyclic antidepressants (amitriptyline)10,23 have been reported to decrease the intensity of pain in a limited number of studies.16,17 Surgical management of FBS also has been attempted by some groups.6,12,13,31,32,34 Netterville et al11 reported excellent results in 1 patient after removal of the auriculotemporal nerve, whereas Lieberman and Har-El34 reported complete resolution of symptoms after total parotidectomy during treatment of a patient with parapharyngeal space malignancy. In addition, neoadjuvant radiotherapy has been reported to aid in resolution of postoperative FBS after neck lymph node dissection by several researchers.6,13,32 In their retrospective cohort study, which reviewed records of 499 patients who underwent surgery of the parapharyngeal space, deep lobe of the parotid gland, and the infratemporal fossa, Linkov et al20 identified an inverse relation between a history of radiation therapy and the development of FBS. However, the authors believe that such a radical approach should be reserved for cases in which radiotherapy is indicated owing to a malignancy, independent of FBS, or for cases in which all other conservative measures have failed. Another approach that has been reported for the management of FBS is intraparotid injection of Botox,4,5,20,29,37,38 although a standard regimen has not been defined.3,4,20 The authors’ experience with this case indicates the need for a treatment approach that should be tailored individually when managing patients with FBS. The present patient was managed conservatively, with improvement of symptoms with medical therapy alone, which the authors recommend as first-line treatment in this subset of patients.
A rare and therefore under-recognized complication of surgery involving the upper neck, FBS also can occur as a complication of alloplastic TMJ replacement. The definitive pathogenesis remains obscure, which supports the theory that the sympathetic and parasympathetic disruption on the myoepithelial cells of the parotid might not be the only contributing factor of FBS. Surgeons performing TMJ replacement procedures should be aware of this potential complication and its outcomes.
References 1. Haubrich WS: The first-bite syndrome. Henry Ford Hosp Med J 34:275, 1986 2. Cernea CR, Hojaij FC, De Carlucci D Jr, et al: First-bite syndrome after resection of the styloid process. Laryngoscope 117:181, 2007 3. Laccourreye O, Werner A, Garcia D, et al: First bite syndrome. Eur Ann Otorhinolaryngol Head Neck Dis 130:269, 2013 4. Ali MJ, Orloff LA, Lustig LR, et al: Botulinum toxin in the treatment of first bite syndrome. Otolaryngol Head Neck Surg 139: 742, 2008 5. Sims JR, Suen JY: First bite syndrome: Case report of three patients treated with botulinum toxin and review of other treatment modalities. Head Neck 35:E288, 2013 6. Chiu AG, Cohen JI, Burningham AR, et al: First bite syndrome: A complication of surgery involving the parapharyngeal space. Head Neck 24:996, 2002 7. Kamal A, Abd El-Fattah AM, Tawfik A, et al: Cervical sympathetic schwannoma with postoperative first bite syndrome. Eur Arch Otorhinolaryngol 264:1109, 2007 8. Costa TP, de Araujo CE, Filipe J, et al: First-bite syndrome in oncologic patients. Eur Arch Otorhinolaryngol 268:1241, 2011 9. Wong EH, Farrier JN, Cooper DG: First-bite syndrome complicating carotid endarterectomy: A case report and literature review. Vasc Endovascular Surg 45:459, 2011 10. Albasri H, Eley KA, Saeed NR: Chronic pain related to first bite syndrome: Report of two cases. Br J Oral Maxillofac Surg 49: 154, 2011 11. Netterville JL, Jackson CG, Miller FR, et al: Vagal paraganglioma: A review of 46 patients treated during a 20-year period. Arch Otolaryngol Head Neck Surg 124:1133, 1998 12. Diercks GR, Rosow DE, Prasad M, et al: A case of preoperative ‘ first-bite syndrome’’ associated with mucoepidermoid carcinoma of the parotid gland. Laryngoscope 121:760, 2011 13. Deganello A, Meccariello G, Busoni M, et al: First bite syndrome as presenting symptom of parapharyngeal adenoid cystic carcinoma. J Laryngol Otol 125:428, 2011 14. Kawashima Y, Sumi T, Sugimoto T, et al: First-bite syndrome: A review of 29 patients with parapharyngeal space tumor. Auris Nasus Larynx 35:109, 2008 15. Fiorini FR, Santoro R, Cristofaro G, et al: Potential use of acupuncture in the treatment of first bite syndrome. Am J Otolaryngol 36:484, 2015 16. Navaie M, Sharghi LH, Cho-Reyes S, et al: Diagnostic approach, treatment, and outcomes of cervical sympathetic chain schwannomas: A global narrative review. Otolaryngol Head Neck Surg 151:899, 2014 17. Ansarin M, Tagliabue M, Chu F, et al: Transoral robotic surgery in retrostyloid parapharyngeal space schwannomas. Case Rep Otolaryngol 2014:296025, 2014 18. Horowitz G, Ben-Ari O, Wasserzug O, et al: The transcervical approach for parapharyngeal space pleomorphic adenomas: Indications and technique. PLoS One 9:e90210, 2014 19. Abdeldaoui A, Oker N, Duet M, et al: First bite syndrome: A little known complication of upper cervical surgery. Eur Ann Otorhinolaryngol Head Neck Dis 130:123, 2013 20. Linkov G, Morris LG, Shah JP, et al: First bite syndrome: Incidence, risk factors, treatment, and outcomes. Laryngoscope 122:1773, 2012
488 21. Chijiwa H, Mihoki T, Shin B, et al: Clinical study of parapharyngeal space tumours. J Laryngol Otol Suppl 31:100, 2009 22. Casserly P, Kiely P, Fenton JE: Cervical sympathetic chain schwannoma masquerading as a carotid body tumour with a postoperative complication of first-bite syndrome. Eur Arch Otorhinolaryngol 266:1659, 2009 23. Phillips TJ, Farquhar-Smith WP: Pharmacological treatment of a patient with first-bite syndrome. Anaesthesia 64:97, 2009 24. Mandel L, Syrop SB: First-bite syndrome after parapharyngeal surgery for cervical schwannoma. J Am Dent Assoc 139:1480, 2008 25. Lin CC, Wang CC, Liu SA, et al: Cervical sympathetic chain schwannoma. J Formos Med Assoc 106:956, 2007 26. Wax MK, Shiley SG, Robinson JL, et al: Cervical sympathetic chain schwannoma. Laryngoscope 114:2210, 2004 27. Smith JJ, Passman MA, Dattilo JB, et al: Carotid body tumor resection: Does the need for vascular reconstruction worsen outcome? Ann Vasc Surg 20:435, 2006 28. Casani AP, Cerchiai N, Dallan I, et al: Benign tumours affecting the deep lobe of the parotid gland: How to select the optimal surgical approach. Acta Otorhinolaryngol Ital 35:80, 2015 29. Mikolajczak S, Ludwig L, Grosheva M, et al: First bite syndrome: Successful treatment with Botulinum toxin A. Laryngorhinootologie 94:924, 2015 (in German)
FIRST BITE SYNDROME AFTER TMJ REPLACEMENT 30. Houle A, Mandel L: First bite syndrome after deep lobe parotidectomy: Case report. J Oral Maxillofac Surg 72:1475, 2014 31. Amin N, Pelser A, Weighill J: First bite syndrome: Our experience of laser tympanic plexus ablation. J Laryngol Otol 128: 166, 2014 32. Guss J, Ashton-Sager AL, Fong BP: First bite syndrome caused by adenoid cystic carcinoma of the submandibular gland. Laryngoscope 123:426, 2013 33. Borras-Perera M, Fortuny-Llanses JC, Palomar-Asenjo V, et al: First-bite syndrome. Acta Otorrinolaringol Esp 60:144, 2009 34. Lieberman SM, Har-El G: First bite syndrome as a presenting symptom of a parapharyngeal space malignancy. Head Neck 33:1539, 2011 35. Scholey AL, Suida MI: First bite syndrome after bimaxillary osteotomy: Case report. Br J Oral Maxillofac Surg 53:561, 2015 36. Sharma PK, Massey BL: Gabapentin for the treatment of first bite syndrome following parapharyngeal space surgery. J Otolaryngol Head Neck Surg 133:P173, 2005 37. Lee BJ, Lee JC, Lee YO, et al: Novel treatment of first bite syndrome using botulinum toxin type A. Head Neck 31:989, 2009 38. Wang TK, Bhamidipaty V, MacCormick M: First bite syndrome following ipsilateral carotid endarterectomy. Vasc Endovascular Surg 47:148, 2013
