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Bilateral lingual anesthesia following surgically assisted rapid palatal expansion: Report of a case
416
BILATERAL LINGUAL ANESTHESIA
14. Saxby MS, Rippin JW, Sheron JE: Case report: Squamous odontogenic tumor of the gingiva. J Periodontol 64:1250, 1993 15. Carr RF, Carlton DM, Marks RB: Squamous odontogenic tumor: Report of a case. J Oral Surg 39:297, 1981 16. Doyle JL, Grodiesh JE, Dolinsky HB: Squamous odontogenic tumor: Report of three cases. J Oral Surg 35:994, 1977 17. McNeil J, Price HM, Stoker NG: Squamous odontogenic tumor: Report of a case with long-term history. J Oral Surg 38:446, 1980 18. Leventon GS, Happonen RP, Newland JR: Squamous odontogenic tumor: Report of two cases and review of the literature. Am J Surg Pathol 5:671, 1981 19. Swan RH, McDaniel RK: Squamous odontogenic proliferation with probable origin from the rests of Malassez (early squamous odontogenic tumor?). J Periodontol 54:493, 1983 20. Tatemoto Y, Okada Y, Mori M: Squamous odontogenic tumor: Immunohistochemical identification of keratins. Oral Surg Oral Med Oral Pathol 67:63, 1989
21. Yaacoob HB: Squamous odontogenic tumor. J Hihon Univ Sch Dent 32:187, 1990 22. Simon JH, Jensen JL: Squamous odontogenic tumor-like proliferations in periapical cysts. J Endod 11:446, 1985 23. Unal T, Gomel M, Gunel O: Squamous odontogenic tumor-like islands in a radicular cyst: Report of a case. J Oral Maxillofac Surg 45:346, 1987 24. Hodgkinson DJ, Woods JE, Dahlin DC, et al: Keratocysts of the jaws. Cancer 41:803, 1978 25. Schwartz-Arad D, Lustmann J, Ulmansky M: Squamous odontogenic tumor. Review of the literature and case report. Int J Oral Maxillofac Surg 19:327, 1990 26. Batsakis JG, Cleary KR: Squamous odontogenic tumor. Ann Otol Rhinol Laryngol 102:823, 1993 27. Haghighat K, Kalmar JR, Mariotti AJ: Squamous odontogenic tumor: Diagnosis and management. J Periodontol 73:653, 2003
J Oral Maxillofac Surg 63:416 – 418, 2005
Bilateral Lingual Anesthesia Following Surgically Assisted Rapid Palatal Expansion: Report of a Case Claris Chuah, DMD,* and Pushkar Mehra, BDS, DMD† Surgically assisted rapid palatal expansion (SARPE) is routinely used to correct maxillary transverse deficiencies in skeletally mature individuals.1 Complications related to the surgical procedure that have been reported in the literature include significant bleeding, injury to branches of the maxillary nerve, infection, pain, devitalization of teeth, periodontal breakdown, relapse, and unilateral expansion.1,2 Complications may also arise due to the expansion appliance, and these include breakage or loosening of the appliance, stripping or locking of the screw, and impingement of the palatal soft tissue causing tissue necrosis.3 A review of the literature does not show any reported cases of lingual nerve injury following SARPE or Le Fort osteotomies. The case presented in this report involves *Resident, Department of Oral and Maxillofacial Surgery, Boston University School of Dental Medicine, Boston, MA. †Director, Department of Oral and Maxillofacial Surgery, Boston Medical Center, and Assistant Professor, Department of Oral and Maxillofacial Surgery, Boston University School of Dental Medicine, Boston, MA. Address correspondence and reprint requests to Dr Mehra: 100 E Newton St, Suite G-407, Boston, MA 02118; e-mail: pushkar. [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6303-0022$30.00/0 doi:10.1016/j.joms.2004.03.021
a patient with bilateral lingual nerve injury after a routine and uncomplicated SARPE procedure.
Report of a Case A 29-year-old white man was referred to the Department of Oral and Maxillofacial Surgery at Boston University Medical Center by his orthodontist for surgical correction of severe maxillary transverse hypoplasia. The patient’s medical history was unremarkable. He was taken to the operating room and intubated under direct laryngoscopy with a nasoendotracheal tube passed through the left nares. The intubation was unremarkable. The surgical technique for the SARPE included the traditional Le Fort I osteotomy cuts, with pterygomaxillary separation with a curved osteotome without maxillary downfracture. Some increased bleeding was visualized bilaterally at the pterygoid plate regions, and this was successfully controlled with Avitene (MedChem products, Woburn, MA) and Surgicel (Ethicon Inc, Somerville, NJ) hemostatic packing. There were no apparent intraoperative complications. The immediate postoperative course was uneventful. There was mild paresthesia in the infraorbital nerve distribution bilaterally, which was not unexpected, and the patient was discharged from the hospital the next day. He was instructed to initiate maxillary expansion on postoperative day 3 at a rate of 0.5-mm expansion twice daily. The patient presented to the emergency department on postoperative day 3 with complaints of weakness and dizziness. Verbal questioning revealed that he had very limited oral intake and had not yet started expansion. Physical examination was unremarkable, and he was treated with intravenous fluid therapy and discharged from the hospital.
CHUAH AND MEHRA He was next seen on a follow-up visit on postoperative day 7. Physical examination revealed slowly resolving bilateral infraorbital paresthesia. He had been expanding the appliance as instructed starting on postoperative day 4. Interestingly, he complained that numbness of his tongue had developed since postoperative day 5. The following objective testing was performed: 1) sharp (pinprick) sensation with a 25-gauge needle, 2) touch sensation with von Frey’s hairs (Semmes-Weinstein monofilaments), 3) cold sensation with ethyl chloride spray, 4) directional sense with camel hair brushstroke, and 5) static and moving 2-point discriminations. This testing showed evidence of complete bilateral anesthesia in the distribution of the lingual nerve. A diagnosis of lingual nerve injury secondary to an unknown etiology was made. The patient was instructed to follow up in 1 week and to continue to expand the appliance as planned. Expansion of the appliance was continued until postoperative day 14 and then stopped as the desired transverse maxillary dimension had been attained. The infraorbital nerve paresthesia had significantly resolved, but the patient reported no improvement with the bilateral lingual nerve anesthesia. Although a detailed examination revealed no changes from the previous examination with respect to testing for thermal sensation, 2-point discrimination, pressure, or direction sense, he was able to subjectively detect pinprick sensation as light touch. An examination on postoperative day 21 showed normal sensation in the infraorbital nerve distribution bilaterally but no changes in the examination of the lingual paresthesia. The patient was instructed to follow up in 2 weeks. The patient reported significant improvement in the bilateral lingual nerve paresthesia on postoperative day 35. He stated that he felt that the “tongue had come alive” in the last 2 to 3 days. Objective testing revealed evidence of almost normal sensation to all testing parameters in the distribution of the lingual nerve bilaterally except the midline dorsal and ventral surfaces of the tongue. Remaining examination was unremarkable. Completely normal tongue sensation was recovered at 87 days after surgery.
Discussion An adequate transverse maxillary dimension is a critical component of a stable and functional occlusion.3 SARPE is a reliable and effective technique for surgical correction of transverse maxillary deficiencies in skeletally mature individuals.4 – 6 Advantages of SARPE over orthodontic therapy and segmental Le Fort procedures include decreased risk of periodontal damage, improved esthetics when smiling, improved nasal air flow, and decreased risk of avascularity, leading to aseptic necrosis. SARPE is also a relatively simple procedure and is associated with minimal morbidity. Intraoperative complications are uncommon, although the risk of bleeding increases if the pterygoid plates are separated from the maxilla.3 Review of the literature did not show any reported cases of lingual nerve injury following SARPE or Le Fort osteotomies. The etiology of the lingual nerve injury in this case remains unclear. The possibility of traumatic injury from a malpositioned oral tongue
417 (Weider) retractor is discarded as this instrument was not kept in the oral cavity during surgery. Although a few cases of lingual nerve injury have been reported after laryngoscopy for intubation,7 this is unlikely in this case as the patient had normal sensation in the tongue for up to 3 days after surgery. The initiation of spontaneous recovery in the fourth week, pattern of gradual improvement, and complete recovery by 3 months after the injury suggests a Sunderland seconddegree nerve injury, which is equivalent to axonotmesis in the Seddon classification.8 –10 We hypothesize that there was an aberration in the anatomic pathway of the lingual nerve in this patient. The injury was bilateral, and this supports the possibility of a variation in the course of the lingual nerve whereby it was in extremely close proximity to the pterygomaxillary region. In our opinion, with the likelihood of the nerve being in an abnormal position, 2 probable mechanisms could have been responsible for this transient injury, as follows: 1. Direct injury to the lingual nerve with the pterygomaxillary osteotomies. The lingual nerve leaves the mandibular nerve and passes downward, forward, and slightly laterally, deep to the lateral pterygoid muscle. It then appears from under the inferior border of the lateral pterygoid muscle and continues downward and forward over the lateral aspect of the medial pterygoid muscle. The lingual nerve then heads toward the lingual aspect of the third molar alveolar region and enters the region of the floor of the mouth.11 The pterygomaxillary osteotomies were performed with a curved osteotome in a standard manner by an experienced surgeon. However, due to the relatively close proximity of the lingual nerve to the pterygoid plates, direct injury to the lingual nerve is certainly a possibility, especially if the course of the nerve was aberrant. 2. Nerve compression from a hematoma in the pterygomaxillary region. Some increased bleeding was encountered intraoperatively, and local hemostatic packing was used bilaterally in the region of the pterygoid plate osteotomies. The most common sources of bleeding in maxillary orthognathic surgery include the terminal branches of the maxillary artery, especially the descending palatine or sphenopalatine arteries, the posterior superior alveolar artery, and the pterygoid venous plexus.1 Surgicel expands in volume before resorption and, although unlikely, could potentially cause nerve compression. Also, expansion of the maxilla itself as performed by the patient could lead to displace-
418
BILATERAL LINGUAL ANESTHESIA
ment of the Surgicel and/or hematoma laterally and cause nerve compression. Minimal consideration has traditionally been given to the protection of the lingual nerve during maxillary orthognathic surgery due to the lack of reports of lingual nerve injury associated with these procedures. Experience with this case suggests that the lingual nerve should be considered as an anatomic structure in close proximity to the surgical region, and patients should be made aware of this potential complication during maxillary orthognathic procedures.
References 1. Mehra P, Cottrell DA, Caiazzo A, et al: Life-threatening, delayed epistaxis after surgically assisted rapid palatal expansion: A case report. J Oral Maxillofac Surg 57:201, 1999
2. Lanigan DT, Hey JH, West RA: Major complications of orthognathic surgery: Hemorrhage associated with Le Fort 1 osteotomies. J Oral Maxillofac Surg 48:561, 1990 3. Silverstein K, Quinn PD: Surgically assisted rapid palatal expansion for the management of transverse maxillary deficiency. J Oral Maxillofac Surg 55:725, 1997 4. Pogrel MA, Kaban LB, Vargervik K, et al: Surgically assisted rapid maxillary expansion in adults. Int J Adult Orthod Orthognath Surg 7:37, 1992 5. Bays RA, Greco JM: Surgically assisted rapid maxillary expansion: An outpatient technique with long term stability. J Oral Maxillofac Surg 50:110, 1992 6. Philips C, Medland WH, Fields HW, et al: Stability of surgical maxillary expansion. Int J Adult Orthod Orthognath Surg 7:139, 1992 7. Lang MS, Waite PD: Bilateral lingual nerve injury after laryngoscopy for intubation. J Oral Maxillofac Surg 59:1497, 2001 8. Sunderlund S: A classification of peripheral nerve injuries producing loss of function. Brain 74:491, 1951 9. Seddon HJ: Three types of nerve injury. Brain 66:237, 1943 10. Fonseca RJ, Walker RV, Betts NJ, et al: Oral and Maxillofacial Trauma, vol 1 (ed 2). Philadelphia, PA, Saunders, 1997 11. Liebgott B: The Anatomical Basis of Dentistry. Philadelphia, PA, BC Decker Inc, 1986, p 304
BILATERAL LINGUAL ANESTHESIA
14. Saxby MS, Rippin JW, Sheron JE: Case report: Squamous odontogenic tumor of the gingiva. J Periodontol 64:1250, 1993 15. Carr RF, Carlton DM, Marks RB: Squamous odontogenic tumor: Report of a case. J Oral Surg 39:297, 1981 16. Doyle JL, Grodiesh JE, Dolinsky HB: Squamous odontogenic tumor: Report of three cases. J Oral Surg 35:994, 1977 17. McNeil J, Price HM, Stoker NG: Squamous odontogenic tumor: Report of a case with long-term history. J Oral Surg 38:446, 1980 18. Leventon GS, Happonen RP, Newland JR: Squamous odontogenic tumor: Report of two cases and review of the literature. Am J Surg Pathol 5:671, 1981 19. Swan RH, McDaniel RK: Squamous odontogenic proliferation with probable origin from the rests of Malassez (early squamous odontogenic tumor?). J Periodontol 54:493, 1983 20. Tatemoto Y, Okada Y, Mori M: Squamous odontogenic tumor: Immunohistochemical identification of keratins. Oral Surg Oral Med Oral Pathol 67:63, 1989
21. Yaacoob HB: Squamous odontogenic tumor. J Hihon Univ Sch Dent 32:187, 1990 22. Simon JH, Jensen JL: Squamous odontogenic tumor-like proliferations in periapical cysts. J Endod 11:446, 1985 23. Unal T, Gomel M, Gunel O: Squamous odontogenic tumor-like islands in a radicular cyst: Report of a case. J Oral Maxillofac Surg 45:346, 1987 24. Hodgkinson DJ, Woods JE, Dahlin DC, et al: Keratocysts of the jaws. Cancer 41:803, 1978 25. Schwartz-Arad D, Lustmann J, Ulmansky M: Squamous odontogenic tumor. Review of the literature and case report. Int J Oral Maxillofac Surg 19:327, 1990 26. Batsakis JG, Cleary KR: Squamous odontogenic tumor. Ann Otol Rhinol Laryngol 102:823, 1993 27. Haghighat K, Kalmar JR, Mariotti AJ: Squamous odontogenic tumor: Diagnosis and management. J Periodontol 73:653, 2003
J Oral Maxillofac Surg 63:416 – 418, 2005
Bilateral Lingual Anesthesia Following Surgically Assisted Rapid Palatal Expansion: Report of a Case Claris Chuah, DMD,* and Pushkar Mehra, BDS, DMD† Surgically assisted rapid palatal expansion (SARPE) is routinely used to correct maxillary transverse deficiencies in skeletally mature individuals.1 Complications related to the surgical procedure that have been reported in the literature include significant bleeding, injury to branches of the maxillary nerve, infection, pain, devitalization of teeth, periodontal breakdown, relapse, and unilateral expansion.1,2 Complications may also arise due to the expansion appliance, and these include breakage or loosening of the appliance, stripping or locking of the screw, and impingement of the palatal soft tissue causing tissue necrosis.3 A review of the literature does not show any reported cases of lingual nerve injury following SARPE or Le Fort osteotomies. The case presented in this report involves *Resident, Department of Oral and Maxillofacial Surgery, Boston University School of Dental Medicine, Boston, MA. †Director, Department of Oral and Maxillofacial Surgery, Boston Medical Center, and Assistant Professor, Department of Oral and Maxillofacial Surgery, Boston University School of Dental Medicine, Boston, MA. Address correspondence and reprint requests to Dr Mehra: 100 E Newton St, Suite G-407, Boston, MA 02118; e-mail: pushkar. [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6303-0022$30.00/0 doi:10.1016/j.joms.2004.03.021
a patient with bilateral lingual nerve injury after a routine and uncomplicated SARPE procedure.
Report of a Case A 29-year-old white man was referred to the Department of Oral and Maxillofacial Surgery at Boston University Medical Center by his orthodontist for surgical correction of severe maxillary transverse hypoplasia. The patient’s medical history was unremarkable. He was taken to the operating room and intubated under direct laryngoscopy with a nasoendotracheal tube passed through the left nares. The intubation was unremarkable. The surgical technique for the SARPE included the traditional Le Fort I osteotomy cuts, with pterygomaxillary separation with a curved osteotome without maxillary downfracture. Some increased bleeding was visualized bilaterally at the pterygoid plate regions, and this was successfully controlled with Avitene (MedChem products, Woburn, MA) and Surgicel (Ethicon Inc, Somerville, NJ) hemostatic packing. There were no apparent intraoperative complications. The immediate postoperative course was uneventful. There was mild paresthesia in the infraorbital nerve distribution bilaterally, which was not unexpected, and the patient was discharged from the hospital the next day. He was instructed to initiate maxillary expansion on postoperative day 3 at a rate of 0.5-mm expansion twice daily. The patient presented to the emergency department on postoperative day 3 with complaints of weakness and dizziness. Verbal questioning revealed that he had very limited oral intake and had not yet started expansion. Physical examination was unremarkable, and he was treated with intravenous fluid therapy and discharged from the hospital.
CHUAH AND MEHRA He was next seen on a follow-up visit on postoperative day 7. Physical examination revealed slowly resolving bilateral infraorbital paresthesia. He had been expanding the appliance as instructed starting on postoperative day 4. Interestingly, he complained that numbness of his tongue had developed since postoperative day 5. The following objective testing was performed: 1) sharp (pinprick) sensation with a 25-gauge needle, 2) touch sensation with von Frey’s hairs (Semmes-Weinstein monofilaments), 3) cold sensation with ethyl chloride spray, 4) directional sense with camel hair brushstroke, and 5) static and moving 2-point discriminations. This testing showed evidence of complete bilateral anesthesia in the distribution of the lingual nerve. A diagnosis of lingual nerve injury secondary to an unknown etiology was made. The patient was instructed to follow up in 1 week and to continue to expand the appliance as planned. Expansion of the appliance was continued until postoperative day 14 and then stopped as the desired transverse maxillary dimension had been attained. The infraorbital nerve paresthesia had significantly resolved, but the patient reported no improvement with the bilateral lingual nerve anesthesia. Although a detailed examination revealed no changes from the previous examination with respect to testing for thermal sensation, 2-point discrimination, pressure, or direction sense, he was able to subjectively detect pinprick sensation as light touch. An examination on postoperative day 21 showed normal sensation in the infraorbital nerve distribution bilaterally but no changes in the examination of the lingual paresthesia. The patient was instructed to follow up in 2 weeks. The patient reported significant improvement in the bilateral lingual nerve paresthesia on postoperative day 35. He stated that he felt that the “tongue had come alive” in the last 2 to 3 days. Objective testing revealed evidence of almost normal sensation to all testing parameters in the distribution of the lingual nerve bilaterally except the midline dorsal and ventral surfaces of the tongue. Remaining examination was unremarkable. Completely normal tongue sensation was recovered at 87 days after surgery.
Discussion An adequate transverse maxillary dimension is a critical component of a stable and functional occlusion.3 SARPE is a reliable and effective technique for surgical correction of transverse maxillary deficiencies in skeletally mature individuals.4 – 6 Advantages of SARPE over orthodontic therapy and segmental Le Fort procedures include decreased risk of periodontal damage, improved esthetics when smiling, improved nasal air flow, and decreased risk of avascularity, leading to aseptic necrosis. SARPE is also a relatively simple procedure and is associated with minimal morbidity. Intraoperative complications are uncommon, although the risk of bleeding increases if the pterygoid plates are separated from the maxilla.3 Review of the literature did not show any reported cases of lingual nerve injury following SARPE or Le Fort osteotomies. The etiology of the lingual nerve injury in this case remains unclear. The possibility of traumatic injury from a malpositioned oral tongue
417 (Weider) retractor is discarded as this instrument was not kept in the oral cavity during surgery. Although a few cases of lingual nerve injury have been reported after laryngoscopy for intubation,7 this is unlikely in this case as the patient had normal sensation in the tongue for up to 3 days after surgery. The initiation of spontaneous recovery in the fourth week, pattern of gradual improvement, and complete recovery by 3 months after the injury suggests a Sunderland seconddegree nerve injury, which is equivalent to axonotmesis in the Seddon classification.8 –10 We hypothesize that there was an aberration in the anatomic pathway of the lingual nerve in this patient. The injury was bilateral, and this supports the possibility of a variation in the course of the lingual nerve whereby it was in extremely close proximity to the pterygomaxillary region. In our opinion, with the likelihood of the nerve being in an abnormal position, 2 probable mechanisms could have been responsible for this transient injury, as follows: 1. Direct injury to the lingual nerve with the pterygomaxillary osteotomies. The lingual nerve leaves the mandibular nerve and passes downward, forward, and slightly laterally, deep to the lateral pterygoid muscle. It then appears from under the inferior border of the lateral pterygoid muscle and continues downward and forward over the lateral aspect of the medial pterygoid muscle. The lingual nerve then heads toward the lingual aspect of the third molar alveolar region and enters the region of the floor of the mouth.11 The pterygomaxillary osteotomies were performed with a curved osteotome in a standard manner by an experienced surgeon. However, due to the relatively close proximity of the lingual nerve to the pterygoid plates, direct injury to the lingual nerve is certainly a possibility, especially if the course of the nerve was aberrant. 2. Nerve compression from a hematoma in the pterygomaxillary region. Some increased bleeding was encountered intraoperatively, and local hemostatic packing was used bilaterally in the region of the pterygoid plate osteotomies. The most common sources of bleeding in maxillary orthognathic surgery include the terminal branches of the maxillary artery, especially the descending palatine or sphenopalatine arteries, the posterior superior alveolar artery, and the pterygoid venous plexus.1 Surgicel expands in volume before resorption and, although unlikely, could potentially cause nerve compression. Also, expansion of the maxilla itself as performed by the patient could lead to displace-
418
BILATERAL LINGUAL ANESTHESIA
ment of the Surgicel and/or hematoma laterally and cause nerve compression. Minimal consideration has traditionally been given to the protection of the lingual nerve during maxillary orthognathic surgery due to the lack of reports of lingual nerve injury associated with these procedures. Experience with this case suggests that the lingual nerve should be considered as an anatomic structure in close proximity to the surgical region, and patients should be made aware of this potential complication during maxillary orthognathic procedures.
References 1. Mehra P, Cottrell DA, Caiazzo A, et al: Life-threatening, delayed epistaxis after surgically assisted rapid palatal expansion: A case report. J Oral Maxillofac Surg 57:201, 1999
2. Lanigan DT, Hey JH, West RA: Major complications of orthognathic surgery: Hemorrhage associated with Le Fort 1 osteotomies. J Oral Maxillofac Surg 48:561, 1990 3. Silverstein K, Quinn PD: Surgically assisted rapid palatal expansion for the management of transverse maxillary deficiency. J Oral Maxillofac Surg 55:725, 1997 4. Pogrel MA, Kaban LB, Vargervik K, et al: Surgically assisted rapid maxillary expansion in adults. Int J Adult Orthod Orthognath Surg 7:37, 1992 5. Bays RA, Greco JM: Surgically assisted rapid maxillary expansion: An outpatient technique with long term stability. J Oral Maxillofac Surg 50:110, 1992 6. Philips C, Medland WH, Fields HW, et al: Stability of surgical maxillary expansion. Int J Adult Orthod Orthognath Surg 7:139, 1992 7. Lang MS, Waite PD: Bilateral lingual nerve injury after laryngoscopy for intubation. J Oral Maxillofac Surg 59:1497, 2001 8. Sunderlund S: A classification of peripheral nerve injuries producing loss of function. Brain 74:491, 1951 9. Seddon HJ: Three types of nerve injury. Brain 66:237, 1943 10. Fonseca RJ, Walker RV, Betts NJ, et al: Oral and Maxillofacial Trauma, vol 1 (ed 2). Philadelphia, PA, Saunders, 1997 11. Liebgott B: The Anatomical Basis of Dentistry. Philadelphia, PA, BC Decker Inc, 1986, p 304