- Email: [email protected]
Intraosseous traumatic neuroma of the maxilla after excision of giant cell granuloma: A case report
1161
SHAH ET AL 3. Kantlas I, Jessurum J, Iamaroon A: Immunohistochemical evaluation and in situ hybridization in a case of oral inverted ductal papilloma. J Oral Maxillofacial Surg 52:503, 1994 4. Marx RE, Stern D: Oral and Maxillofacial Pathology. Carol Stream, IL, Quintessence Publishing Co, 2003 5. Hegarty DJ, Hopper C, Speight PM: Inverted ductal papilloma of minor salivary glands. J Oral Pathol Med 23:334, 1994 6. Neville BW, Damin OD, Allen CM: Oral and Maxillofacial Pathology. Philadelphia, PA, Saunders, 1995, pp 347-349 7. Woodson GE, Robbins KT, Michaels L: Inverted papillomas, considerations in treatment. Arch Otolaryngol 111:806, 1985 8. Ulrich HP, Mohr C, Schettler D, et al: Inverted papilloma: Implications for the oral and maxillofacial surgeon. Dtsch Z Mund Kiefer Gesichtschir 14:165, 1990 9. Weissler MC, Montgomery WW, Turner PA, et al: Inverted papilloma. Ann Otol Rhinol Laryngol 95:215, 1986 10. Augulo JC, Lopez JI, Toledo JD, et al: Inverted papillary carcinoma. Arch Esp Urol 46:695, 1993 11. Takagi M, Ishikawa G: Benign papillary squamous tumors of the mouth. Bull Tokyo Med Dent Univ 29:17, 1982
12. Boesen PV, Laszewski MJ, Robinson RA, et al: Squamous cell carcinoma in an inverted papilloma of the buccal mucosa. Ann Otol Rhinol Laryngol 100:748, 1991 13. Lesperance MM, Esclamado RM: Squamous cell carcinoma arising in inverted papilloma. Laryngoscope 105:178, 1995 14. Lawson W, Ho B, Shaari C, et al: Inverted papilloma: A report of 112 cases. Laryngoscope 105:282, 1995 15. Hegarty DJ, Hopper C, Speight PM: Inverted ductal papilloma of minor salivary glands. J Oral Pathol Med 23:334, 1994 16. Brannin RB, Sciubba JJ, Giulani M: Ductal papillomas of salivary gland origin, a report of 19 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 92:68, 2001 17. White DK, Miller AS, McDaniel RK, et al: Inverted ductal papilloma. Cancer 49:519, 1982 18. Ellis GL, AuClair PL: Tumors of the Salivary Glands—Ductal Papillomas. Philadelphia, PA, Saunders, 1991, p 238 19. Clark DB, Priddy RW, Swanson AE: Oral inverted ductal papilloma. Oral Surg Oral Med Oral Pathol 69:487, 1990 20. Wilson DF, Robinson BW: Oral inverted ductal papilloma. Oral Surg Oral Med Oral Pathol 57:520, 1984 J Oral Maxillofac Surg 62:1161-1164, 2004
Intraosseous Traumatic Neuroma of the Maxilla After Excision of Giant Cell Granuloma: A Case Report Sonal S. Shah, DDS,* Julien Ghannoum, DMD,† Alan Carness, DDS,‡ and Paul D. Freedman, DDS§ Traumatic neuromas are typically found within the oral soft tissues following surgery, dental extractions, accidental trauma, or trauma from parafunctional habits. The most common intraoral sites are the mental foramen area, lower lip, and tongue.1,2 Intraosseous lesions of the jawbones are rare, and those reported have almost exclusively been within the mandible.2,3 Sist and Greene2 suggested that neuromas may not readily develop in bone because of pressure from the surrounding tissues. Centrally located traumatic neuromas can occur following many types of mandibular
*Resident, Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. †Former Resident, Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. ‡Private Practice, Oral and Maxillofacial Surgery, New York, NY. §Director, Section of Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. Address correspondence and reprint requests to Dr Shah: Oral Pathology Laboratory, Inc, 57-20 141st St, Flushing, NY 11355; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6209-0023$30.00/0 doi:10.1016/j.joms.2003.11.016
surgeries, especially sagittal split osteotomy of the ramus.1 In these cases, they occur as a consequence of interruption of the inferior alveolar nerve or a branch of it, such as the mental nerve. However, traumatic neuromas of the maxilla are rare. To our knowledge, only one other such case has been reported in the English literature. In 1971, Lee described a case of amputation neuroma of the maxilla.4 We report another case of this rare entity in this article. Traumatic neuromas are the body’s attempt to repair an injured nerve and thus are not true tumors. A proliferation of neuronal tissue occurs following damage to a nerve. Microscopically, numerous haphazardly arranged small nerve trunks, cut longitudinally and transversely, can be seen in a background of dense fibrous connective tissue and viable bone trabeculae, if found in bone. The lesion is usually nonencapsulated.
Report of a Case In 1993, a 57-year-old woman presented for biopsy after a large multilocular radiolucency extending from the right maxillary second premolar area to the left maxillary lateral incisor area was discovered on a panoramic radiograph (Fig
1162
INTRAOSSEOUS TRAUMATIC NEUROMA OF THE MAXILLA
FIGURE 2. Computed tomography scan exhibiting a radiolucency in the right maxillary region. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
were also seen. These histologic observations were consistent with the diagnosis of traumatic neuroma.
FIGURE 1. Panoramic radiograph of the original lesion, diagnosed as central giant cell granuloma. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
1). Although the patient was asymptomatic, there was significant expansion of the buccal cortex of the right maxilla. An incisional bone biopsy was performed and sent for surgical pathology consultation. On surgical removal, the lesion was solid, brown in color, and bled easily. The biopsy specimen consisted of multiple pieces of tan soft tissue measuring 1.3 ⫻ 1.0 ⫻ 0.7 cm. Microscopic examination revealed clusters of foreign body–type giant cells set in a well-vascularized, hemorrhagic, fibroblastic, loosely arranged stroma. Lymphocytes, plasma cells, and osteoid trabeculae were also noted. Based on the histologic appearance, a diagnosis of central giant cell granuloma was rendered. Because the histology of this lesion is indistinguishable from the giant cell lesion of hyperparathyroidism, medical evaluation to rule out hyperparathyroidism was indicated. The patient was evaluated for hyperparathyroidism. Serum studies for calcium, phosphorus, and mid-range parathormone were performed. The patient was in the normal range for each of these. The giant cell lesion was then completely excised, debrided, and curetted from the right maxilla under general anesthesia in the operating room. During the surgical procedure, multiple perforations of the maxillary buccal cortical plate by the lesion were noted. The patient was followed yearly after excision. The patient was asymptomatic and no recurrences were detected. In March 2002, panoramic radiographs, computed tomography scans (Fig 2), and occlusal x-rays (Fig 3) demonstrated the presence of a radiolucency located in the right maxilla in the area of the previous lesion. These findings were incidental because the patient was asymptomatic with an unremarkable intraoral clinical appearance. A recurrence was suspected and consequently an excisional biopsy was performed. Intraoperatively, the lesion was found to extend between the right lateral wall of the right nostril and the right medial wall of the antrum. The mass was found to perforate into the antrum and had the clinical appearance of fibrous tissue. The specimen consisted of 3 pieces of tan soft tissue measuring 1.0 ⫻ 0.6 ⫻ 0.2 cm. Microscopically, dense fibrous connective tissue containing numerous haphazardly arranged small nerve trunks was observed (Fig 4). Regional mucous gland lobules and viable bone trabeculae
Discussion Most traumatic neuromas that occur in the oral cavity present as painful nodules or lumps in the soft tissue; their occurrence in bone is much less common. Our case was unique in that not only was the lesion intraosseous, but it occurred in the maxilla rather than the mandible. The mandible is a much more likely site of intraosseous neuromas because the inferior alveolar nerve and its many branches course directly through the mandibular bone. Furthermore,
FIGURE 3. Occlusal radiograph demonstrating a radiolucency in the right maxilla. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
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FIGURE 4. Medium-power photomicrograph showing numerous small nerve trunks in a fibrous background, consistent with the diagnosis of traumatic neuroma (Hematoxylin-eosin stain; magnification ⫻ 10). Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
the mandible is a site of major oral surgeries such as the sagittal split osteotomy. The histologic findings of extensive proliferative and regenerative response of nerve tissue can be attributed to trauma caused by previous surgical intervention. In our case, a maxillary nerve was most likely injured during excision of the large central giant cell granuloma. Injury to a branch of the superior alveolar or one of the palatine nerves is the most likely etiology. The majority of traumatic neuromas are painless and asymptomatic. Women account for 75% of the patients that report pain associated with neuromas.5 The differential diagnosis for traumatic neuromas of the head and neck includes schwannomas and neurofibromas. The standard treatment is complete surgical excision. The prognosis is good and recurrence is rare.6 Many theories have been proposed to describe the pathogenesis of traumatic neuromas. In 1805, the English physiologist Waller put forth his theory of nerve degeneration and regeneration. He stated that the nerve axis cylinder becomes fibrillated and disintegrates and the nerve sheath breaks up into droplets of myelin. Schwann cells are then converted to phagocytes to remove the remnants of the nerve sheath and the axis cylinder. Following degeneration of the proximal nerve ends, the reparative process begins to occur. Axons, Schwann cells, endoneurium, and perineurium proliferate and grow into the injured area. The axon will continue to regenerate in an attempt to reach the endoneurium of the distal stump. However, if this distance is too great, the axis cylinder may coil up and form a nodule covered by fibrous tissue. This nodule consists of neuronal tissue in a fibrous tissue background and is thus referred to as a neuroma.4
In 1985, Badalamente et al7 claimed that ultrastructural alcian blue staining and biochemical analyses revealed a glucosamine-glycosaminoglycan matrix within neuromas when compared with control nerves. Electron microscopic examination revealed that fibroblasts play a major role in the pathogenesis of human neuromas. They are responsible for the proliferation of collagen that occurs after nerve injury. Myofibroblasts also play a significant part in neuroma formation. These cells appear to be involved in the scarring response that occurs after nerve injury; however, their origin is uncertain. Myofibroblasts were not detected in samples of intact nerves. In 1998, Macias et al8 conducted studies to determine the possible existence of regeneration differences between axon subtypes. They concluded that sensory axons regenerated at a faster rate than motor axons, but motor axons regenerated to a greater quantity. They also stated that the distance from the injury site to the neuronal cell body significantly affects regeneration; the greater this distance, the lesser the possibility of successful repair and regeneration. In 2001, Kryger et al9 claimed that nerve growth factor might play a role in the pathogenesis of neuroma formation as well as in the development of neuropathic pain. Their research found that inhibiting nerve growth factor following peripheral nerve injury in the rat reduces neuroma formation and neuropathic pain. In 2002, Xu and Zochodne10 examined the characteristics of long-term experimental neuromas created by sciatic nerve transection. They determined that the microvascular environment created in the proximal nerve stump at the site of injury influences the potential for nerve regrowth and development of neuropathic pain. Their studies suggested that neuroma formation is related to ischemia, which impedes regeneration. They found that local blood flow and perfusion declined in the distal stump of the severed nerve to values considerably lower than observed in intact nerves. In conclusion, intraosseous traumatic neuromas are rare, but probably occur much more frequently than are reported. When a lesion reappears after surgical removal of a tumor in the same anatomic area, traumatic neuroma should always be included in the differential diagnosis. However, a definitive biopsy should be performed to rule out recurrent disease.
References 1. Chau MY, Jonsson E, Lee KM: Traumatic neuroma following sagittal mandibular osteotomy. Int J Oral Maxillofac Surg 18:95, 1989
1164
TWO-STAGE DISTRACTION OSTEOGENESIS
2. Sist TC, Greene GW: Traumatic neuroma of the oral cavity. Oral Surg Oral Med Oral Pathol 51:394, 1981 3. Zain RB, Ling KC: Traumatic neuroma in wall of recurrent unicystic ameloblastoma: A case report. Med J Malaysia 40:49, 1985 4. Lee HT: Amputation neuroma of the maxilla. Dent J Malaysia Singapore 11:17, 1971 5. Cawson RA, Binnie WH, Speight PM, et al: Lucas’s Pathology of Tumors of the Oral Tissues (ed 5). New York, NY, Churchill Livingstone, 1998 6. Lee EJ, Calcaterra TC, Zuckerbraun L: Traumatic neuromas of the head and neck. Ear Nose Throat J 77:670, 1998
7. Badalamente MA, Hurst LC, Ellstein J, et al: The pathobiology of human neuromas: An electron microscopic and biochemical study. J Hand Surg [Am] 10:49, 1985 8. Macias MY, Lehman CT, Sanger JR, et al: Myelinated sensory and alpha motor axon regeneration in peripheral nerve neuromas. Muscle Nerve 21:1748, 1998 9. Kryger GS, Kryger Z, Zhang F, et al: Nerve growth factor inhibition prevents traumatic neuroma formation in the rat. J Hand Surg [Am] 26:635, 2001 10. Xu QG, Zochodne DW: Ischemia and failed regeneration in chronic experimental neuromas. Brain Res 946:24, 2002
J Oral Maxillofac Surg 62:1164-1168, 2004
Two-Stage Distraction Osteogenesis for Mandibular Segmental Defect Masayuki Fukuda, DDS, PhD,* Mitsuyoshi Iino, DDS, PhD,† Kaoru Yamaoka, DDS,‡ Takayoshi Ohnuki, DDS,§ Hirokazu Nagai, DDS, PhD,㛳 and Tetsu Takahashi, DDS, PhD¶ Over the past 10 years distraction osteogenesis has become increasingly popular and has been applied to maxillofacial region for many cases.1-3 Recently, osteogenesis with the transport or compression method has been used for mandibular reconstruction of segmental bone defects.4-8 Although each distraction method is well established for repairing extremities, there is, to our knowledge, no report of combined use of both the transport and compression method for mandibular segmental bone defects. This report presents a case of 2-stage distraction osteogenesis followed by implant therapy for mandibular reconstruction after segmental osteotomy. *Lecturer, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. †Lecturer, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. ‡Senior Resident, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. §Assistant Professor, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. 㛳Assistant Professor, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. ¶Professor and Chairman, Second Department of Oral and Maxillofacial Surgery, Kyushu Dental College, Kitakyushu, Japan. Address correspondence and reprint requests to Dr Fukuda: Division of Dentistry and Oral Surgery, Akita University School of Medicine, 1-1-1 Hondo, Akita 010-8543, Japan; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6209-0024$30.00/0 doi:10.1016/j.joms.2003.11.017
Report of a Case A 61-year-old man was referred to our division for swelling of the left mandibular body. A panoramic radiograph and computed tomography scan showed a radiolucency from the right canine to the left second premolar region of the mandible. Microscopic examination of the biopsy specimen revealed ameloblastoma of the mandible. The initial treatment plan was to perform mandibular segmental ostectomy and bone grafting for reconstruction. However, the patient refused to undergo bone grafting. Therefore, we planned distraction osteogenesis for mandibular reconstruction. Under general anesthesia, 55 mm of the mandibular body was resected by segmental ostectomy intraorally and extraorally. Immediate reconstruction with a titanium reconstruction plate was performed (Figs 1, 2). Because the precise 3-dimensional reconstruction for the curved mandibular defect with the use of ready-made distractors was quite difficult, we planned 2-stage distraction osteogenesis (ie, the transport method first followed by the compression method).5-7 Eight months later, we performed transport method (ie, bifocal distraction osteogenesis) for the defect. With the patient under general anesthesia, the titanium plate was changed, and an internal mandibular distraction device (horizontal type; Medicon eG, Tuttlingen, Germany) was adjusted and preplated before osteotomy. Subperiosteal corticotomy for the transport segment was then performed with a reciprocating saw between the second premolar tooth and the first molar tooth on the right side. The distraction device was fixed to both the transport segment and the basal bone (Fig 3). The function of the distractor was then checked for any bony interferences. The surgical incision was closed, leaving part of the distractor passing through the intraoral incision. Eleven days after surgery, distraction was performed at a rate of 0.8 mm per day (0.4 mm in the morning and 0.4 mm in the evening). The gain obtained in 25 days was 20 mm in the horizontal direction (Figs 4, 5).
SHAH ET AL 3. Kantlas I, Jessurum J, Iamaroon A: Immunohistochemical evaluation and in situ hybridization in a case of oral inverted ductal papilloma. J Oral Maxillofacial Surg 52:503, 1994 4. Marx RE, Stern D: Oral and Maxillofacial Pathology. Carol Stream, IL, Quintessence Publishing Co, 2003 5. Hegarty DJ, Hopper C, Speight PM: Inverted ductal papilloma of minor salivary glands. J Oral Pathol Med 23:334, 1994 6. Neville BW, Damin OD, Allen CM: Oral and Maxillofacial Pathology. Philadelphia, PA, Saunders, 1995, pp 347-349 7. Woodson GE, Robbins KT, Michaels L: Inverted papillomas, considerations in treatment. Arch Otolaryngol 111:806, 1985 8. Ulrich HP, Mohr C, Schettler D, et al: Inverted papilloma: Implications for the oral and maxillofacial surgeon. Dtsch Z Mund Kiefer Gesichtschir 14:165, 1990 9. Weissler MC, Montgomery WW, Turner PA, et al: Inverted papilloma. Ann Otol Rhinol Laryngol 95:215, 1986 10. Augulo JC, Lopez JI, Toledo JD, et al: Inverted papillary carcinoma. Arch Esp Urol 46:695, 1993 11. Takagi M, Ishikawa G: Benign papillary squamous tumors of the mouth. Bull Tokyo Med Dent Univ 29:17, 1982
12. Boesen PV, Laszewski MJ, Robinson RA, et al: Squamous cell carcinoma in an inverted papilloma of the buccal mucosa. Ann Otol Rhinol Laryngol 100:748, 1991 13. Lesperance MM, Esclamado RM: Squamous cell carcinoma arising in inverted papilloma. Laryngoscope 105:178, 1995 14. Lawson W, Ho B, Shaari C, et al: Inverted papilloma: A report of 112 cases. Laryngoscope 105:282, 1995 15. Hegarty DJ, Hopper C, Speight PM: Inverted ductal papilloma of minor salivary glands. J Oral Pathol Med 23:334, 1994 16. Brannin RB, Sciubba JJ, Giulani M: Ductal papillomas of salivary gland origin, a report of 19 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 92:68, 2001 17. White DK, Miller AS, McDaniel RK, et al: Inverted ductal papilloma. Cancer 49:519, 1982 18. Ellis GL, AuClair PL: Tumors of the Salivary Glands—Ductal Papillomas. Philadelphia, PA, Saunders, 1991, p 238 19. Clark DB, Priddy RW, Swanson AE: Oral inverted ductal papilloma. Oral Surg Oral Med Oral Pathol 69:487, 1990 20. Wilson DF, Robinson BW: Oral inverted ductal papilloma. Oral Surg Oral Med Oral Pathol 57:520, 1984 J Oral Maxillofac Surg 62:1161-1164, 2004
Intraosseous Traumatic Neuroma of the Maxilla After Excision of Giant Cell Granuloma: A Case Report Sonal S. Shah, DDS,* Julien Ghannoum, DMD,† Alan Carness, DDS,‡ and Paul D. Freedman, DDS§ Traumatic neuromas are typically found within the oral soft tissues following surgery, dental extractions, accidental trauma, or trauma from parafunctional habits. The most common intraoral sites are the mental foramen area, lower lip, and tongue.1,2 Intraosseous lesions of the jawbones are rare, and those reported have almost exclusively been within the mandible.2,3 Sist and Greene2 suggested that neuromas may not readily develop in bone because of pressure from the surrounding tissues. Centrally located traumatic neuromas can occur following many types of mandibular
*Resident, Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. †Former Resident, Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. ‡Private Practice, Oral and Maxillofacial Surgery, New York, NY. §Director, Section of Oral and Maxillofacial Pathology, New York Hospital Queens, Flushing, NY. Address correspondence and reprint requests to Dr Shah: Oral Pathology Laboratory, Inc, 57-20 141st St, Flushing, NY 11355; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6209-0023$30.00/0 doi:10.1016/j.joms.2003.11.016
surgeries, especially sagittal split osteotomy of the ramus.1 In these cases, they occur as a consequence of interruption of the inferior alveolar nerve or a branch of it, such as the mental nerve. However, traumatic neuromas of the maxilla are rare. To our knowledge, only one other such case has been reported in the English literature. In 1971, Lee described a case of amputation neuroma of the maxilla.4 We report another case of this rare entity in this article. Traumatic neuromas are the body’s attempt to repair an injured nerve and thus are not true tumors. A proliferation of neuronal tissue occurs following damage to a nerve. Microscopically, numerous haphazardly arranged small nerve trunks, cut longitudinally and transversely, can be seen in a background of dense fibrous connective tissue and viable bone trabeculae, if found in bone. The lesion is usually nonencapsulated.
Report of a Case In 1993, a 57-year-old woman presented for biopsy after a large multilocular radiolucency extending from the right maxillary second premolar area to the left maxillary lateral incisor area was discovered on a panoramic radiograph (Fig
1162
INTRAOSSEOUS TRAUMATIC NEUROMA OF THE MAXILLA
FIGURE 2. Computed tomography scan exhibiting a radiolucency in the right maxillary region. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
were also seen. These histologic observations were consistent with the diagnosis of traumatic neuroma.
FIGURE 1. Panoramic radiograph of the original lesion, diagnosed as central giant cell granuloma. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
1). Although the patient was asymptomatic, there was significant expansion of the buccal cortex of the right maxilla. An incisional bone biopsy was performed and sent for surgical pathology consultation. On surgical removal, the lesion was solid, brown in color, and bled easily. The biopsy specimen consisted of multiple pieces of tan soft tissue measuring 1.3 ⫻ 1.0 ⫻ 0.7 cm. Microscopic examination revealed clusters of foreign body–type giant cells set in a well-vascularized, hemorrhagic, fibroblastic, loosely arranged stroma. Lymphocytes, plasma cells, and osteoid trabeculae were also noted. Based on the histologic appearance, a diagnosis of central giant cell granuloma was rendered. Because the histology of this lesion is indistinguishable from the giant cell lesion of hyperparathyroidism, medical evaluation to rule out hyperparathyroidism was indicated. The patient was evaluated for hyperparathyroidism. Serum studies for calcium, phosphorus, and mid-range parathormone were performed. The patient was in the normal range for each of these. The giant cell lesion was then completely excised, debrided, and curetted from the right maxilla under general anesthesia in the operating room. During the surgical procedure, multiple perforations of the maxillary buccal cortical plate by the lesion were noted. The patient was followed yearly after excision. The patient was asymptomatic and no recurrences were detected. In March 2002, panoramic radiographs, computed tomography scans (Fig 2), and occlusal x-rays (Fig 3) demonstrated the presence of a radiolucency located in the right maxilla in the area of the previous lesion. These findings were incidental because the patient was asymptomatic with an unremarkable intraoral clinical appearance. A recurrence was suspected and consequently an excisional biopsy was performed. Intraoperatively, the lesion was found to extend between the right lateral wall of the right nostril and the right medial wall of the antrum. The mass was found to perforate into the antrum and had the clinical appearance of fibrous tissue. The specimen consisted of 3 pieces of tan soft tissue measuring 1.0 ⫻ 0.6 ⫻ 0.2 cm. Microscopically, dense fibrous connective tissue containing numerous haphazardly arranged small nerve trunks was observed (Fig 4). Regional mucous gland lobules and viable bone trabeculae
Discussion Most traumatic neuromas that occur in the oral cavity present as painful nodules or lumps in the soft tissue; their occurrence in bone is much less common. Our case was unique in that not only was the lesion intraosseous, but it occurred in the maxilla rather than the mandible. The mandible is a much more likely site of intraosseous neuromas because the inferior alveolar nerve and its many branches course directly through the mandibular bone. Furthermore,
FIGURE 3. Occlusal radiograph demonstrating a radiolucency in the right maxilla. Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
1163
SHAH ET AL
FIGURE 4. Medium-power photomicrograph showing numerous small nerve trunks in a fibrous background, consistent with the diagnosis of traumatic neuroma (Hematoxylin-eosin stain; magnification ⫻ 10). Shah et al. Intraosseous Traumatic Neuroma of the Maxilla. J Oral Maxillofac Surg 2004.
the mandible is a site of major oral surgeries such as the sagittal split osteotomy. The histologic findings of extensive proliferative and regenerative response of nerve tissue can be attributed to trauma caused by previous surgical intervention. In our case, a maxillary nerve was most likely injured during excision of the large central giant cell granuloma. Injury to a branch of the superior alveolar or one of the palatine nerves is the most likely etiology. The majority of traumatic neuromas are painless and asymptomatic. Women account for 75% of the patients that report pain associated with neuromas.5 The differential diagnosis for traumatic neuromas of the head and neck includes schwannomas and neurofibromas. The standard treatment is complete surgical excision. The prognosis is good and recurrence is rare.6 Many theories have been proposed to describe the pathogenesis of traumatic neuromas. In 1805, the English physiologist Waller put forth his theory of nerve degeneration and regeneration. He stated that the nerve axis cylinder becomes fibrillated and disintegrates and the nerve sheath breaks up into droplets of myelin. Schwann cells are then converted to phagocytes to remove the remnants of the nerve sheath and the axis cylinder. Following degeneration of the proximal nerve ends, the reparative process begins to occur. Axons, Schwann cells, endoneurium, and perineurium proliferate and grow into the injured area. The axon will continue to regenerate in an attempt to reach the endoneurium of the distal stump. However, if this distance is too great, the axis cylinder may coil up and form a nodule covered by fibrous tissue. This nodule consists of neuronal tissue in a fibrous tissue background and is thus referred to as a neuroma.4
In 1985, Badalamente et al7 claimed that ultrastructural alcian blue staining and biochemical analyses revealed a glucosamine-glycosaminoglycan matrix within neuromas when compared with control nerves. Electron microscopic examination revealed that fibroblasts play a major role in the pathogenesis of human neuromas. They are responsible for the proliferation of collagen that occurs after nerve injury. Myofibroblasts also play a significant part in neuroma formation. These cells appear to be involved in the scarring response that occurs after nerve injury; however, their origin is uncertain. Myofibroblasts were not detected in samples of intact nerves. In 1998, Macias et al8 conducted studies to determine the possible existence of regeneration differences between axon subtypes. They concluded that sensory axons regenerated at a faster rate than motor axons, but motor axons regenerated to a greater quantity. They also stated that the distance from the injury site to the neuronal cell body significantly affects regeneration; the greater this distance, the lesser the possibility of successful repair and regeneration. In 2001, Kryger et al9 claimed that nerve growth factor might play a role in the pathogenesis of neuroma formation as well as in the development of neuropathic pain. Their research found that inhibiting nerve growth factor following peripheral nerve injury in the rat reduces neuroma formation and neuropathic pain. In 2002, Xu and Zochodne10 examined the characteristics of long-term experimental neuromas created by sciatic nerve transection. They determined that the microvascular environment created in the proximal nerve stump at the site of injury influences the potential for nerve regrowth and development of neuropathic pain. Their studies suggested that neuroma formation is related to ischemia, which impedes regeneration. They found that local blood flow and perfusion declined in the distal stump of the severed nerve to values considerably lower than observed in intact nerves. In conclusion, intraosseous traumatic neuromas are rare, but probably occur much more frequently than are reported. When a lesion reappears after surgical removal of a tumor in the same anatomic area, traumatic neuroma should always be included in the differential diagnosis. However, a definitive biopsy should be performed to rule out recurrent disease.
References 1. Chau MY, Jonsson E, Lee KM: Traumatic neuroma following sagittal mandibular osteotomy. Int J Oral Maxillofac Surg 18:95, 1989
1164
TWO-STAGE DISTRACTION OSTEOGENESIS
2. Sist TC, Greene GW: Traumatic neuroma of the oral cavity. Oral Surg Oral Med Oral Pathol 51:394, 1981 3. Zain RB, Ling KC: Traumatic neuroma in wall of recurrent unicystic ameloblastoma: A case report. Med J Malaysia 40:49, 1985 4. Lee HT: Amputation neuroma of the maxilla. Dent J Malaysia Singapore 11:17, 1971 5. Cawson RA, Binnie WH, Speight PM, et al: Lucas’s Pathology of Tumors of the Oral Tissues (ed 5). New York, NY, Churchill Livingstone, 1998 6. Lee EJ, Calcaterra TC, Zuckerbraun L: Traumatic neuromas of the head and neck. Ear Nose Throat J 77:670, 1998
7. Badalamente MA, Hurst LC, Ellstein J, et al: The pathobiology of human neuromas: An electron microscopic and biochemical study. J Hand Surg [Am] 10:49, 1985 8. Macias MY, Lehman CT, Sanger JR, et al: Myelinated sensory and alpha motor axon regeneration in peripheral nerve neuromas. Muscle Nerve 21:1748, 1998 9. Kryger GS, Kryger Z, Zhang F, et al: Nerve growth factor inhibition prevents traumatic neuroma formation in the rat. J Hand Surg [Am] 26:635, 2001 10. Xu QG, Zochodne DW: Ischemia and failed regeneration in chronic experimental neuromas. Brain Res 946:24, 2002
J Oral Maxillofac Surg 62:1164-1168, 2004
Two-Stage Distraction Osteogenesis for Mandibular Segmental Defect Masayuki Fukuda, DDS, PhD,* Mitsuyoshi Iino, DDS, PhD,† Kaoru Yamaoka, DDS,‡ Takayoshi Ohnuki, DDS,§ Hirokazu Nagai, DDS, PhD,㛳 and Tetsu Takahashi, DDS, PhD¶ Over the past 10 years distraction osteogenesis has become increasingly popular and has been applied to maxillofacial region for many cases.1-3 Recently, osteogenesis with the transport or compression method has been used for mandibular reconstruction of segmental bone defects.4-8 Although each distraction method is well established for repairing extremities, there is, to our knowledge, no report of combined use of both the transport and compression method for mandibular segmental bone defects. This report presents a case of 2-stage distraction osteogenesis followed by implant therapy for mandibular reconstruction after segmental osteotomy. *Lecturer, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. †Lecturer, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. ‡Senior Resident, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. §Assistant Professor, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. 㛳Assistant Professor, Division of Dentistry and Oral Surgery, Akita University School of Medicine, Akita, Japan. ¶Professor and Chairman, Second Department of Oral and Maxillofacial Surgery, Kyushu Dental College, Kitakyushu, Japan. Address correspondence and reprint requests to Dr Fukuda: Division of Dentistry and Oral Surgery, Akita University School of Medicine, 1-1-1 Hondo, Akita 010-8543, Japan; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6209-0024$30.00/0 doi:10.1016/j.joms.2003.11.017
Report of a Case A 61-year-old man was referred to our division for swelling of the left mandibular body. A panoramic radiograph and computed tomography scan showed a radiolucency from the right canine to the left second premolar region of the mandible. Microscopic examination of the biopsy specimen revealed ameloblastoma of the mandible. The initial treatment plan was to perform mandibular segmental ostectomy and bone grafting for reconstruction. However, the patient refused to undergo bone grafting. Therefore, we planned distraction osteogenesis for mandibular reconstruction. Under general anesthesia, 55 mm of the mandibular body was resected by segmental ostectomy intraorally and extraorally. Immediate reconstruction with a titanium reconstruction plate was performed (Figs 1, 2). Because the precise 3-dimensional reconstruction for the curved mandibular defect with the use of ready-made distractors was quite difficult, we planned 2-stage distraction osteogenesis (ie, the transport method first followed by the compression method).5-7 Eight months later, we performed transport method (ie, bifocal distraction osteogenesis) for the defect. With the patient under general anesthesia, the titanium plate was changed, and an internal mandibular distraction device (horizontal type; Medicon eG, Tuttlingen, Germany) was adjusted and preplated before osteotomy. Subperiosteal corticotomy for the transport segment was then performed with a reciprocating saw between the second premolar tooth and the first molar tooth on the right side. The distraction device was fixed to both the transport segment and the basal bone (Fig 3). The function of the distractor was then checked for any bony interferences. The surgical incision was closed, leaving part of the distractor passing through the intraoral incision. Eleven days after surgery, distraction was performed at a rate of 0.8 mm per day (0.4 mm in the morning and 0.4 mm in the evening). The gain obtained in 25 days was 20 mm in the horizontal direction (Figs 4, 5).