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Botulinum toxin in the treatment of first bite syndrome
Otolaryngology–Head and Neck Surgery (2008) 139, 742-743
CASE REPORT
Botulinum toxin in the treatment of first bite syndrome M. Jafer Ali, MD, Lisa A. Orloff, MD, Lawrence R. Lustig, MD, and David W. Eisele, MD, San Francisco, CA
F
irst bite syndrome (FBS) following surgery of the parapharyngeal space is a well known but poorly understood complication.1 Most theories on the pathology of FBS revolve around the concept of sympathetic denervation of the parotid gland with subsequent hypersensitivity of myoepithelial cells to parasympathetic neurotransmitters. This hypersensitivity is thought to elicit a supramaximal contraction of myoepithelial cells during the first bite of a meal that subsides with continued masticatory action.2 Although the intricacies of FBS pathways may not be fully understood, the concept that pain is elicited ultimately with myoepithelial contraction led us to hypothesize that paralysis of these myoepithelial filaments with botulinum toxin may result in relief of symptoms associated with FBS. We report the first documented use of botulinum toxin in the treatment of FBS. This case report was approved by the University of California, San Francisco Committee on Human Research (IRB). The patient is a 53-year-old woman with a history of right neck lymphangioma. She had undergone four operations for surgical resection in 1984, 2004, 2005, and 2006. In 2005, resection included mandibular osteotomies and parapharyngeal space dissection (Fig 1). After this operation, she developed a Horner’s syndrome and complaints of intense radiating pain in the preauricular region upon the first bite of each meal that improved with subsequent masticatory movements. She was given the diagnosis of first bite syndrome and was treated medically with aggressive pain management including multiple narcotics and neurotrophic drugs. She also tried therapeutic acupuncture but gained little relief from her symptoms. She elected to undergo tympanic neurectomy in 2007, which initially provided her with what she described as 50% relief from her symptoms. However, much of her pain returned over the next few months and she elected to undergo a second tympanic neurectomy with laser ablation of the promontory six months later, which provided no further relief of symptoms. Four months later, she was evaluated in our office and continued to have severe pain with first bite. A lemon drop
Figure 1 Exposure of parapharyngeal space with double mandibular osteotomies.
was given to the patient during consultation that resulted in 10/10 intense pain in the preauricular parotid region. This continued for 10 minutes, then resolved on its own. The patient reported that these symptoms were consistent with her usual symptoms with first bite at meals. The patient agreed to undergo a trial of botulinum toxin treatment in our office. Under ultrasound visualization, 75 units of botulinum toxin diluted into 2 mL of saline solution was injected into the right parotid gland diffusely and with focus on areas where the patient felt the most intense pain (Fig 2). The injection resulted in immediate pain and pressure upon infusion that resolved completely within five minutes. Less than 48 hours later, the patient reported near-complete resolution of her first bite symptoms. At 10-week follow-up, she continues to have no further symptoms, with a pain score of 0/10. Furthermore, she reports a dramatic improvement in quality of life. The patient will be followed clinically. If the effect of botulinum toxin is similar to that of patients treated for sialorrhea, then it is likely that her pain will recur within
Received June 12, 2008; revised August 7, 2008; accepted August 13, 2008.
0194-5998/$34.00 © 2008 American Academy of Otolaryngology–Head and Neck Surgery Foundation. All rights reserved. doi:10.1016/j.otohns.2008.08.015
Ali et al
Botulinum toxin in the treatment of first . . .
743 resent a novel primary treatment for FBS, given its relatively minimal morbidity. Further studies are needed to better understand its efficacy and longevity.
AUTHOR INFORMATION From the Department of Otolaryngology–Head and Neck Surgery, University of California, San Francisco. Corresponding author: M. Jafer Ali, MD, Department of Otolaryngology– Head and Neck Surgery, University of California, San Francisco, 400 Parnassus Ave-ACC 7th Floor, Box 0342, San Francisco, CA 94143-0342.
Figure 2 Ultrasound-guided botulinum toxin injection of right parotid gland.
four to six months. At that time, she would be offered a repeat injection.
AUTHOR CONTRIBUTIONS M. Jafer Ali, MD, primary author/primary investigator; Lisa A. Orloff, MD, investigator/surgeon/ultrasound; Lawrence R. Lustig, MD, investigator/surgeon; David W. Eisele, MD, investigator/surgeon.
FINANCIAL DISCLOSURE DISCUSSION We report a novel treatment for FBS associated with parapharyngeal space surgery. Botulinum toxin has been used for a number disease entities relating to the salivary glands including gustatory sweating3 after parotidectomy and sialorrhea in patients with neurological disorders.4 The latter likely involves a mechanism whereby botulinum toxin blocks presynaptic acetylcholine release, which renders myoepithelial cells ineffective. The end result in this case is to decrease salivary flow. In FBS, the paralysis of myoepithelial cells may inhibit supramaximal contraction of hypersensitized myoepithelial cells and, as a result, alleviate pain symptoms. Although we report a case of medically and surgically refractory FBS, we propose that botulinum toxin may rep-
None.
REFERENCES 1. Cohen SM, Burkey BB, Netterville JL. Surgical management of parapharyngeal space masses. Head Neck 2005;27(8):669 –75. 2. Kawashima Y, Sumi T, Sugimoto T, et al. First-bite syndrome: a review of 29 patients with parapharyngeal space tumor. Auris Nasus Larynx 2008;35(1):109 –13. 3. Ferraro G, Altieri A, Grella E, et al. Botulinum toxin: 28 patients affected by Frey’s syndrome treated with intradermal injections. Plast Reconstr Surg 2005;115(1):344 –5. 4. Porta M, Gamba M, Bertacchi G, et al. Treatment of sialorrhea with ultrasound guided botulinum toxin type A injection in patients with neurological disabilities. J Neurol Neurosurg Psychiatr 2001;70(4): 538 – 40.
CASE REPORT
Botulinum toxin in the treatment of first bite syndrome M. Jafer Ali, MD, Lisa A. Orloff, MD, Lawrence R. Lustig, MD, and David W. Eisele, MD, San Francisco, CA
F
irst bite syndrome (FBS) following surgery of the parapharyngeal space is a well known but poorly understood complication.1 Most theories on the pathology of FBS revolve around the concept of sympathetic denervation of the parotid gland with subsequent hypersensitivity of myoepithelial cells to parasympathetic neurotransmitters. This hypersensitivity is thought to elicit a supramaximal contraction of myoepithelial cells during the first bite of a meal that subsides with continued masticatory action.2 Although the intricacies of FBS pathways may not be fully understood, the concept that pain is elicited ultimately with myoepithelial contraction led us to hypothesize that paralysis of these myoepithelial filaments with botulinum toxin may result in relief of symptoms associated with FBS. We report the first documented use of botulinum toxin in the treatment of FBS. This case report was approved by the University of California, San Francisco Committee on Human Research (IRB). The patient is a 53-year-old woman with a history of right neck lymphangioma. She had undergone four operations for surgical resection in 1984, 2004, 2005, and 2006. In 2005, resection included mandibular osteotomies and parapharyngeal space dissection (Fig 1). After this operation, she developed a Horner’s syndrome and complaints of intense radiating pain in the preauricular region upon the first bite of each meal that improved with subsequent masticatory movements. She was given the diagnosis of first bite syndrome and was treated medically with aggressive pain management including multiple narcotics and neurotrophic drugs. She also tried therapeutic acupuncture but gained little relief from her symptoms. She elected to undergo tympanic neurectomy in 2007, which initially provided her with what she described as 50% relief from her symptoms. However, much of her pain returned over the next few months and she elected to undergo a second tympanic neurectomy with laser ablation of the promontory six months later, which provided no further relief of symptoms. Four months later, she was evaluated in our office and continued to have severe pain with first bite. A lemon drop
Figure 1 Exposure of parapharyngeal space with double mandibular osteotomies.
was given to the patient during consultation that resulted in 10/10 intense pain in the preauricular parotid region. This continued for 10 minutes, then resolved on its own. The patient reported that these symptoms were consistent with her usual symptoms with first bite at meals. The patient agreed to undergo a trial of botulinum toxin treatment in our office. Under ultrasound visualization, 75 units of botulinum toxin diluted into 2 mL of saline solution was injected into the right parotid gland diffusely and with focus on areas where the patient felt the most intense pain (Fig 2). The injection resulted in immediate pain and pressure upon infusion that resolved completely within five minutes. Less than 48 hours later, the patient reported near-complete resolution of her first bite symptoms. At 10-week follow-up, she continues to have no further symptoms, with a pain score of 0/10. Furthermore, she reports a dramatic improvement in quality of life. The patient will be followed clinically. If the effect of botulinum toxin is similar to that of patients treated for sialorrhea, then it is likely that her pain will recur within
Received June 12, 2008; revised August 7, 2008; accepted August 13, 2008.
0194-5998/$34.00 © 2008 American Academy of Otolaryngology–Head and Neck Surgery Foundation. All rights reserved. doi:10.1016/j.otohns.2008.08.015
Ali et al
Botulinum toxin in the treatment of first . . .
743 resent a novel primary treatment for FBS, given its relatively minimal morbidity. Further studies are needed to better understand its efficacy and longevity.
AUTHOR INFORMATION From the Department of Otolaryngology–Head and Neck Surgery, University of California, San Francisco. Corresponding author: M. Jafer Ali, MD, Department of Otolaryngology– Head and Neck Surgery, University of California, San Francisco, 400 Parnassus Ave-ACC 7th Floor, Box 0342, San Francisco, CA 94143-0342.
Figure 2 Ultrasound-guided botulinum toxin injection of right parotid gland.
four to six months. At that time, she would be offered a repeat injection.
AUTHOR CONTRIBUTIONS M. Jafer Ali, MD, primary author/primary investigator; Lisa A. Orloff, MD, investigator/surgeon/ultrasound; Lawrence R. Lustig, MD, investigator/surgeon; David W. Eisele, MD, investigator/surgeon.
FINANCIAL DISCLOSURE DISCUSSION We report a novel treatment for FBS associated with parapharyngeal space surgery. Botulinum toxin has been used for a number disease entities relating to the salivary glands including gustatory sweating3 after parotidectomy and sialorrhea in patients with neurological disorders.4 The latter likely involves a mechanism whereby botulinum toxin blocks presynaptic acetylcholine release, which renders myoepithelial cells ineffective. The end result in this case is to decrease salivary flow. In FBS, the paralysis of myoepithelial cells may inhibit supramaximal contraction of hypersensitized myoepithelial cells and, as a result, alleviate pain symptoms. Although we report a case of medically and surgically refractory FBS, we propose that botulinum toxin may rep-
None.
REFERENCES 1. Cohen SM, Burkey BB, Netterville JL. Surgical management of parapharyngeal space masses. Head Neck 2005;27(8):669 –75. 2. Kawashima Y, Sumi T, Sugimoto T, et al. First-bite syndrome: a review of 29 patients with parapharyngeal space tumor. Auris Nasus Larynx 2008;35(1):109 –13. 3. Ferraro G, Altieri A, Grella E, et al. Botulinum toxin: 28 patients affected by Frey’s syndrome treated with intradermal injections. Plast Reconstr Surg 2005;115(1):344 –5. 4. Porta M, Gamba M, Bertacchi G, et al. Treatment of sialorrhea with ultrasound guided botulinum toxin type A injection in patients with neurological disabilities. J Neurol Neurosurg Psychiatr 2001;70(4): 538 – 40.