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Partial resection of the mandible for the treatment of diffuse sclerosing osteomyelitis: Report of four cases
414
DISCUSSION
including the lingual and buccal cortices and teeth in the lesion, but the histologic specimensfrom the resection margins showed that the inflammatory change was present. Although operative removal of pathologic tissue has produced good results in the case of suppurative osteomyelitis,9’10in DSO casesit may be necessary to resect the mandible more widely beyond the abnormal area detected radiographically and surgically. Under this constraint, esthetic disturbance and disability of oral function will become more severe. We therefore believe that mandibular resection should be limited to severe, therapy-resistant cases, as Montonen at al2 pointed out, and that the extent of the resection should be established more carefully, using other procedures such as technetium bone scan, computed tomography, and magnetic resonanceimaging before the operation. Regarding the cause of DSO, Wannfors4 concluded after the study of chronic osteomyelitis that bacteria were never found in vital bone, and inflammation was probably not maintained by the actual spreadof microorganismsbut by the releaseof bacterial toxins through the mandibular bone. If this hypothesis is correct and DSO is caused by bacterial infection, just as other types of osteomyelitis, our patients would have been cured with partial resection because the initial symptomatic focus was completely removed and the resected margin showed vital bone histologically. Furthermore, in patient 3, the resection margin did not show any inflammatory change. Conversely, the direct spread of bacteria in the bone marrow and extension of the osteomyelitic lesion would also conflict with our observation that grafted iliac bone was not involved by the lesion even though it was attached to the mandible in which the lesion recurred and spread. The mandible and the grafted iliac bone differ in that the former possessesnervous and periosteal interfaces. We con-
sider that these structures may be related to the occurrence or extension of the DSO lesion. Although we cannot confirm the cause of DSO, we agree that only direct bacteriologic infection of the mandibular bone should not be postulated as the cause of this otherwise inexplicable disease. Acknowledgment The authors thank Dr Nobuhiko Kono and Dr Akihiro Kanagawa for kindly providing the data on patient 1.
References 1. Marx RE, Carlson ER, Smith BR, et al: Isolation of actinomyces species and eikenella corrodens from patients with chronic diffuse sclerosing osteomyelitis. J Oral Maxillofac Surg 52:26, 1994 2. Montonen M, Iizuka T, Hallikainen D, et al: Decortication in the treatment of diffuse sclerosing osteomyelitis of the mandible: Retrospective analysis of 41 cases between 1969 and 1990. Oral Surg Oral Med Oral Path01 75:5, 1993 3. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: A new concept of its etiology. Oral Surg Oral Med Oral Path01 70:414, 1990 4. Wannfors K: Chronic osteomyelitis of the jaws (thesis). Stockholm: Karolinska Institutet, 1990, pp 4-56 5. van Merkesteyn JPR, Groot RH, Bras .I, et al: Diffuse sclerosing osteomyelitis of the mandible: Clinical radiographic and histologic findings in twenty-seven patients. J Oral Maxillofac Surg 46:825, 1988 6. Jacobsson S: Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg 13:363, 1984 7. Malmstrijm M, Fyhrquist F, Kosunen TU, et al: Immunological features of patients with chronic sclerosing osteomyelitis of the mandible. Int J Oral Surg 12:6, 1983 8. Jacobsson S, Hollender L: Treatment and prognosis of diffuse sclerosing osteomyelitis (DSO) of the mandible. Oral Surg Oral Med Oral Path01 49:7, 1980 9. Hjorting-Hansen E: Decortication in treatment of osteomyelitis of the mandible. Oral Surg Oral Med Oral Path01 29:641, 1970 10. Obwegeser HL, Sailer HF: Experiences with intra-oral partial resection and simultaneous reconstruction in cases of mandibular osteomyelitis. J Maxillofac Surg 6:34, 1978
J Oral Maxillofac Surg 55:414-415. 1997
Uiscussion Partial Resection of the Mandible for the Treatment of Diffuse Sclerosing Osteomyelitis: Report of Four Cases J.P.R. van Merkesteyn, DOS, PhD Academic
Hospital
Leiden,
Leiden,
The Netherlands
In the well documented report of Dr Suei et al on “Partial Resection of the Mandible for the Treatment of Diffuse Sclerosing Osteomyelitis - Report on Four Cases,” diffuse sclerosing osteomyelitis (DSO) of the mandible is considered a well established entity of which the etiology has not yet been clarified. Although a brief reference is made to two reports
of the work of van Merkesteyn, et al. the reviewedliterature on the etiology of DSO lacks referenceto further work on the hypothesisthat DSO of the mandibleis not an osteomyelitis but rather represents a reactive hyperplasia of bone secondary to a chronic periostitis caused by muscular overuse.‘.’ The so-called “diffuse sclerosing osteomyelitis” was extensively reviewed in 1984 by Jacobsson. As Suei et al state, this lesion is characterized by recurrent pain and/or swelling and trismus without suppuration or fistula forma-
tion. Antibiotic treatment was usually ineffective and the etiology hasbeenobscurefor many years. One of the major problems with this lesion has been the fact that although the patients showed several symptoms of
BEZIAT ET AL
415
osteomyelitis,the histology never showedsignsof active osteomyelitis.The histology is characterizedby remodeling of cortical bone, subperiostealformation of new bone, and a subcorticalincreaseof trabecularbonevolume. The stroma is scanty to moderately fibrous, well vascularized, and shows,whenpresent,few inflammatorycells.‘%2.6 Thesefindingsareconsistentthroughoutthe literature andfit a reactive lesion of bone rather than an active osteomyelitis.In the presentreport of Sueiet al this mostimportantfinding isonce again illustrated,especiallyby their report of fatty marrow without inflammation in patient 3 (Fig 4B), in a part of the mandiblewhererecurrent diseaseis found later on. The radiographic appearanceof the lesion is characterizedby diffuse sclerosisor intermingled sclerosisand osteolysis. Subperiostalbone formation (1, 2) is found in most cases and, more important, the lesion seemsto occur in different locations than suppurativeosteomyelitis.‘.’These findings are alsoillustratedin the casesof Dr. Suei et al. In their discussionregardingthe etiology of DSO Suei et al conclude that from their material an indirect bacterial cause(spreadof bacterial toxins) or direct bacterial spread in the bonemarrow is not likely. They even statethat “the mandible and grafted iliac bone differ in that the former possesses nervousandperiostealinterfacesandconcludethat “these structuresmay be related to the occurrenceand/or extensionof the DSO lesion.” Consideringthe finding of involvement of the condyles in three of the four patients, the histopathologicfindings,the fact that the bonegraft was not affected by the recurrent lesion, and the fact that the
attachmentof the bonegraft wasnot affected,it seemslikely that the causeof the bonechangesis not locatedin the bone itself but at the periostealinterface. Thus the reported four cases instead of being examples of an “inexplicable disease,” seemto give further well documentedevidencesup-
porting the hypothesisthat DSO is not an osteomyelitisbut rather it represents a reactive hyperplasia of bone as the result of a chronic periostitis caused by muscular overuse.
References 1. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: Clinical, radiographic and histopathological findings in 27 patients. J Oral Maxillofac Surg 46:825, 1988 2. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: A new concept of its etiology. Oral Surg Oral Med Oral Path01 70:414, 1990 3. Groot RH, van Merkesteyn JPR, van Soest JJ et al: Diffuse sclerosing osteomyelitis (chronic tendoperiostitis) of the mandible: An 1l-year follow-up report. Oral Surg Oral Med Oral Path01 74:557, 1992 4. Groot RH, Ongerboer de Visser BW, van Merkesteyn JPR, et al: Changes in inhibitory reflex responses in patients with diffuse sclerosing osteomyelitis of the mandible. Oral Surg Oral Med Oral Path01 74:727, 1992 5. Groot RH, van Merkesteyn JPR, Bras J: Diffuse sclerosing osteomyelitis and florid osseous dysplasia. Oral Surg Oral Med Oral Path01 81:333, 1996 6. Jacobsson S: Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg 13:363, 1984
J Oral Maxillofac Surg 55:415-419, 1997
Central Vascular Malformation of the Mandible: A Case Report JEAN-LUC BEZIAT, MD,* JOAO-PEDRO MARCELINO, MDT YVES BASCOULERGUE, MD* AND DANIELLE VITREY, MD5 Hemangiomas of bone are uncommon intraosseous lesions consisting of a proliferation of blood vessels. The central hemangioma occurs most frequently in the vertebrae and the skull (0.2% of osseousneoplasms).’ * Professor of Maxillofacial Surgery, Service de Chirurgie MaxIlo-Faciale, C.H.U. Lyon Nord, HGpital de la Croix-Rousse, Lyon, France. t Resident, Service de Chirurgie Maxillo-Faciale, C.H.U. Lyon Nord, HBpital de la Croix-Rousse, Lyon, France. $ In Private Practice, Service de Radiologie, Clinique de la Sauvegarde, Lyon, France. 5 Clinical Associate Doctor, Laboratoire d’Anatomie Pathologie, Lyon, France. -Address correspondence and reprint requests to Dr Beziat: Service de Chirurgie Maxillo-Faciale, C.H.U. Lvon Nord, HBpital de la Croix-Rouise, 96, Grande Rue de la Cr&Rousse, 69‘004 Lyon, France. 0 1997 American Association 0278-2391/97/5504-0016$3.00/O
of Oral and Maxillofacial
Surgeons
The mandible and the maxilla are the next most common sitesof occurrence. The causeof central hemangiomas is unknown. Some lesions may represent true neoplasms; others are more probably developmental or traumatic in origin.’ More than half of the central hemangiomas of the jaws occur in the mandible, with the posterior region being the most frequent site.2’3The lesion occurs approximately twice as frequently in women as in men. The peak age of incidence is the second decade of life.2%4 A slow-growing, asymmetric expansion of the mandible or maxilla is the most common complaint. Spontaneous gingival bleeding around teeth in the area may also be noted. Paresthesiaor pain is occasionally evident, as well as mobility of involved teeth. The teeth may exhibit a pumping action so that, when depressed
DISCUSSION
including the lingual and buccal cortices and teeth in the lesion, but the histologic specimensfrom the resection margins showed that the inflammatory change was present. Although operative removal of pathologic tissue has produced good results in the case of suppurative osteomyelitis,9’10in DSO casesit may be necessary to resect the mandible more widely beyond the abnormal area detected radiographically and surgically. Under this constraint, esthetic disturbance and disability of oral function will become more severe. We therefore believe that mandibular resection should be limited to severe, therapy-resistant cases, as Montonen at al2 pointed out, and that the extent of the resection should be established more carefully, using other procedures such as technetium bone scan, computed tomography, and magnetic resonanceimaging before the operation. Regarding the cause of DSO, Wannfors4 concluded after the study of chronic osteomyelitis that bacteria were never found in vital bone, and inflammation was probably not maintained by the actual spreadof microorganismsbut by the releaseof bacterial toxins through the mandibular bone. If this hypothesis is correct and DSO is caused by bacterial infection, just as other types of osteomyelitis, our patients would have been cured with partial resection because the initial symptomatic focus was completely removed and the resected margin showed vital bone histologically. Furthermore, in patient 3, the resection margin did not show any inflammatory change. Conversely, the direct spread of bacteria in the bone marrow and extension of the osteomyelitic lesion would also conflict with our observation that grafted iliac bone was not involved by the lesion even though it was attached to the mandible in which the lesion recurred and spread. The mandible and the grafted iliac bone differ in that the former possessesnervous and periosteal interfaces. We con-
sider that these structures may be related to the occurrence or extension of the DSO lesion. Although we cannot confirm the cause of DSO, we agree that only direct bacteriologic infection of the mandibular bone should not be postulated as the cause of this otherwise inexplicable disease. Acknowledgment The authors thank Dr Nobuhiko Kono and Dr Akihiro Kanagawa for kindly providing the data on patient 1.
References 1. Marx RE, Carlson ER, Smith BR, et al: Isolation of actinomyces species and eikenella corrodens from patients with chronic diffuse sclerosing osteomyelitis. J Oral Maxillofac Surg 52:26, 1994 2. Montonen M, Iizuka T, Hallikainen D, et al: Decortication in the treatment of diffuse sclerosing osteomyelitis of the mandible: Retrospective analysis of 41 cases between 1969 and 1990. Oral Surg Oral Med Oral Path01 75:5, 1993 3. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: A new concept of its etiology. Oral Surg Oral Med Oral Path01 70:414, 1990 4. Wannfors K: Chronic osteomyelitis of the jaws (thesis). Stockholm: Karolinska Institutet, 1990, pp 4-56 5. van Merkesteyn JPR, Groot RH, Bras .I, et al: Diffuse sclerosing osteomyelitis of the mandible: Clinical radiographic and histologic findings in twenty-seven patients. J Oral Maxillofac Surg 46:825, 1988 6. Jacobsson S: Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg 13:363, 1984 7. Malmstrijm M, Fyhrquist F, Kosunen TU, et al: Immunological features of patients with chronic sclerosing osteomyelitis of the mandible. Int J Oral Surg 12:6, 1983 8. Jacobsson S, Hollender L: Treatment and prognosis of diffuse sclerosing osteomyelitis (DSO) of the mandible. Oral Surg Oral Med Oral Path01 49:7, 1980 9. Hjorting-Hansen E: Decortication in treatment of osteomyelitis of the mandible. Oral Surg Oral Med Oral Path01 29:641, 1970 10. Obwegeser HL, Sailer HF: Experiences with intra-oral partial resection and simultaneous reconstruction in cases of mandibular osteomyelitis. J Maxillofac Surg 6:34, 1978
J Oral Maxillofac Surg 55:414-415. 1997
Uiscussion Partial Resection of the Mandible for the Treatment of Diffuse Sclerosing Osteomyelitis: Report of Four Cases J.P.R. van Merkesteyn, DOS, PhD Academic
Hospital
Leiden,
Leiden,
The Netherlands
In the well documented report of Dr Suei et al on “Partial Resection of the Mandible for the Treatment of Diffuse Sclerosing Osteomyelitis - Report on Four Cases,” diffuse sclerosing osteomyelitis (DSO) of the mandible is considered a well established entity of which the etiology has not yet been clarified. Although a brief reference is made to two reports
of the work of van Merkesteyn, et al. the reviewedliterature on the etiology of DSO lacks referenceto further work on the hypothesisthat DSO of the mandibleis not an osteomyelitis but rather represents a reactive hyperplasia of bone secondary to a chronic periostitis caused by muscular overuse.‘.’ The so-called “diffuse sclerosing osteomyelitis” was extensively reviewed in 1984 by Jacobsson. As Suei et al state, this lesion is characterized by recurrent pain and/or swelling and trismus without suppuration or fistula forma-
tion. Antibiotic treatment was usually ineffective and the etiology hasbeenobscurefor many years. One of the major problems with this lesion has been the fact that although the patients showed several symptoms of
BEZIAT ET AL
415
osteomyelitis,the histology never showedsignsof active osteomyelitis.The histology is characterizedby remodeling of cortical bone, subperiostealformation of new bone, and a subcorticalincreaseof trabecularbonevolume. The stroma is scanty to moderately fibrous, well vascularized, and shows,whenpresent,few inflammatorycells.‘%2.6 Thesefindingsareconsistentthroughoutthe literature andfit a reactive lesion of bone rather than an active osteomyelitis.In the presentreport of Sueiet al this mostimportantfinding isonce again illustrated,especiallyby their report of fatty marrow without inflammation in patient 3 (Fig 4B), in a part of the mandiblewhererecurrent diseaseis found later on. The radiographic appearanceof the lesion is characterizedby diffuse sclerosisor intermingled sclerosisand osteolysis. Subperiostalbone formation (1, 2) is found in most cases and, more important, the lesion seemsto occur in different locations than suppurativeosteomyelitis.‘.’These findings are alsoillustratedin the casesof Dr. Suei et al. In their discussionregardingthe etiology of DSO Suei et al conclude that from their material an indirect bacterial cause(spreadof bacterial toxins) or direct bacterial spread in the bonemarrow is not likely. They even statethat “the mandible and grafted iliac bone differ in that the former possesses nervousandperiostealinterfacesandconcludethat “these structuresmay be related to the occurrenceand/or extensionof the DSO lesion.” Consideringthe finding of involvement of the condyles in three of the four patients, the histopathologicfindings,the fact that the bonegraft was not affected by the recurrent lesion, and the fact that the
attachmentof the bonegraft wasnot affected,it seemslikely that the causeof the bonechangesis not locatedin the bone itself but at the periostealinterface. Thus the reported four cases instead of being examples of an “inexplicable disease,” seemto give further well documentedevidencesup-
porting the hypothesisthat DSO is not an osteomyelitisbut rather it represents a reactive hyperplasia of bone as the result of a chronic periostitis caused by muscular overuse.
References 1. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: Clinical, radiographic and histopathological findings in 27 patients. J Oral Maxillofac Surg 46:825, 1988 2. van Merkesteyn JPR, Groot RH, Bras J, et al: Diffuse sclerosing osteomyelitis of the mandible: A new concept of its etiology. Oral Surg Oral Med Oral Path01 70:414, 1990 3. Groot RH, van Merkesteyn JPR, van Soest JJ et al: Diffuse sclerosing osteomyelitis (chronic tendoperiostitis) of the mandible: An 1l-year follow-up report. Oral Surg Oral Med Oral Path01 74:557, 1992 4. Groot RH, Ongerboer de Visser BW, van Merkesteyn JPR, et al: Changes in inhibitory reflex responses in patients with diffuse sclerosing osteomyelitis of the mandible. Oral Surg Oral Med Oral Path01 74:727, 1992 5. Groot RH, van Merkesteyn JPR, Bras J: Diffuse sclerosing osteomyelitis and florid osseous dysplasia. Oral Surg Oral Med Oral Path01 81:333, 1996 6. Jacobsson S: Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg 13:363, 1984
J Oral Maxillofac Surg 55:415-419, 1997
Central Vascular Malformation of the Mandible: A Case Report JEAN-LUC BEZIAT, MD,* JOAO-PEDRO MARCELINO, MDT YVES BASCOULERGUE, MD* AND DANIELLE VITREY, MD5 Hemangiomas of bone are uncommon intraosseous lesions consisting of a proliferation of blood vessels. The central hemangioma occurs most frequently in the vertebrae and the skull (0.2% of osseousneoplasms).’ * Professor of Maxillofacial Surgery, Service de Chirurgie MaxIlo-Faciale, C.H.U. Lyon Nord, HGpital de la Croix-Rousse, Lyon, France. t Resident, Service de Chirurgie Maxillo-Faciale, C.H.U. Lyon Nord, HBpital de la Croix-Rousse, Lyon, France. $ In Private Practice, Service de Radiologie, Clinique de la Sauvegarde, Lyon, France. 5 Clinical Associate Doctor, Laboratoire d’Anatomie Pathologie, Lyon, France. -Address correspondence and reprint requests to Dr Beziat: Service de Chirurgie Maxillo-Faciale, C.H.U. Lvon Nord, HBpital de la Croix-Rouise, 96, Grande Rue de la Cr&Rousse, 69‘004 Lyon, France. 0 1997 American Association 0278-2391/97/5504-0016$3.00/O
of Oral and Maxillofacial
Surgeons
The mandible and the maxilla are the next most common sitesof occurrence. The causeof central hemangiomas is unknown. Some lesions may represent true neoplasms; others are more probably developmental or traumatic in origin.’ More than half of the central hemangiomas of the jaws occur in the mandible, with the posterior region being the most frequent site.2’3The lesion occurs approximately twice as frequently in women as in men. The peak age of incidence is the second decade of life.2%4 A slow-growing, asymmetric expansion of the mandible or maxilla is the most common complaint. Spontaneous gingival bleeding around teeth in the area may also be noted. Paresthesiaor pain is occasionally evident, as well as mobility of involved teeth. The teeth may exhibit a pumping action so that, when depressed