A case of mandibular hemophilic pseudotumor

A hemophilic pseudotumor was found in the mandible a of a 13-year-old boy with a known history of moderate hemophilia A. Intraorally, the alveolus was enlarged buccally and lingually in the left retromolar region. Hypesthesia of the left mental region was noted. Panoramic X-ray film revealed a lytic...

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Published inJapanese Journal of Oral and Maxillofacial Surgery Vol. 46; no. 2; pp. 105 - 107
Main Authors SHIROZU, Takamasa, MIYACHI, Hitoshi, KAWAHARA, Kou, HOTTA, Fumio, TOHYA, Tousei
Format Journal Article
LanguageEnglish
Published Japanese Society of Oral and Maxillofacial Surgeons 2000
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ISSN0021-5163
2186-1579
DOI10.5794/jjoms.46.105

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Abstract A hemophilic pseudotumor was found in the mandible a of a 13-year-old boy with a known history of moderate hemophilia A. Intraorally, the alveolus was enlarged buccally and lingually in the left retromolar region. Hypesthesia of the left mental region was noted. Panoramic X-ray film revealed a lytic bone lesion from 7 to the left angle of the mandible. Computed tomography revealed marked enlargement of the left side of the body of the mandible, associated with a low density area, and irregular absorption of the lingual cortex. A malignant tumor was suspected, so a biopsy was done during replacement therapy with coagulation factor (Factor VIII). The bone cavity contained a hematoma and was surrounded by granulation-like tissue. A histopathologic diagnosis of hemophilic pseudotumor was made. One year after curretage, the patient's condition was normal, both clinically and radiologically. The pathogenetic mechanism was hypothesized to be a pressure-necrosis phenomenon due to intraosseous bleeding.
AbstractList A hemophilic pseudotumor was found in the mandible a of a 13-year-old boy with a known history of moderate hemophilia A. Intraorally, the alveolus was enlarged buccally and lingually in the left retromolar region. Hypesthesia of the left mental region was noted. Panoramic X-ray film revealed a lytic bone lesion from 7 to the left angle of the mandible. Computed tomography revealed marked enlargement of the left side of the body of the mandible, associated with a low density area, and irregular absorption of the lingual cortex. A malignant tumor was suspected, so a biopsy was done during replacement therapy with coagulation factor (Factor VIII). The bone cavity contained a hematoma and was surrounded by granulation-like tissue. A histopathologic diagnosis of hemophilic pseudotumor was made. One year after curretage, the patient's condition was normal, both clinically and radiologically. The pathogenetic mechanism was hypothesized to be a pressure-necrosis phenomenon due to intraosseous bleeding.
Author KAWAHARA, Kou
SHIROZU, Takamasa
MIYACHI, Hitoshi
HOTTA, Fumio
TOHYA, Tousei
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  organization: Department of Dentistry and Oral and Maxillofacial Surgery, Gifu Prefectural Tajimi Hospital
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  organization: Department of Dentistry and Oral and Maxillofacial Surgery, Gifu Prefectural Tajimi Hospital
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  fullname: TOHYA, Tousei
  organization: Department of Dentistry and Oral and Maxillofacial Surgery, Gifu Prefectural Tajimi Hospital
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References 9) 工藤正文, 横山浩之, 他: 血友病性偽腫瘍の1例. 八戸市民病院医誌12: 16-19 1989.
11) Sousa, S. M., Piratininga, J. L., et al.: Hemophilic pseudotumor of the jaws: report of a cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 79: 216-219 1995.
2) Stoneman, D. W. and Beierl, C. D.: Pseudotumor of hemophilia in mandible. Oral Surg Oral Med Oral Pathol 40: 811-815 1975.
12) Zheng, K. and Zheng, P.: Hemophilic pseudotumor involving maxilla and tibia. Chin Med J 110: 233-235 1997.
3) Mulkey, T. F.: Hemophilic pseudotumor of the mandible. J Oral Surg 35: 561-568 1977.
7) Correra, A., Buckley, J., et al.: Radiotherapy of a pseudotumor in a hemophiliac with factor Mil inhibitor. Am J Pediatr Hematol Oncol 6: 325-327 1984.
10) Bryan, G. W., Leibold, D. G., et al.: Hemophilic pseudotumor of the mandible. Oral Surg Oral Med Oral Pathol 69: 550-553 1990.
14) Abell, J. M. and Bailey R. W.: Hemophilic pseudotumor: two cases occuring in sublings. Arch Surg 81: 569-581 1960.
5) Marquets, J. L., Vinageras, E., et al.: Hemophilic pseudotumor of the inferior maxilla. Oral Surg Oral Med Oral Pathol 53: 347-350 1982.
6) Martinez-Lage, J. L., Sanchez, H., et al.: A pseudotumor of the mandible in a haemophiliac patient. J Maxillofac Surg 11: 171-173 1983.
13) 川中正雄, 柳澤高道, 他: 幼児に生じた下顎骨血友病性偽腫瘍の1例. 口科誌46: 414-418 1997.
1) Lazorovitis, P. and Greim, M. L.: Radiotherapy of haemophilia pseudotumors. Radiology 91: 1026-1027 1968.
8) Brook, A. H., Bedi, R., et al.: Hemophilic pseudotumor of the mandible: report of a case in a one-year-old child. Br J Oral Maxillofac Surg 23: 47-52 1985.
4) 志村俊晴, 長尾寿, 他: 上顎骨中に埋伏歯を伴う血友病性血液嚢胞の手術治験例について. 交通医学33: 103-109 1979.
References_xml – reference: 1) Lazorovitis, P. and Greim, M. L.: Radiotherapy of haemophilia pseudotumors. Radiology 91: 1026-1027 1968.
– reference: 10) Bryan, G. W., Leibold, D. G., et al.: Hemophilic pseudotumor of the mandible. Oral Surg Oral Med Oral Pathol 69: 550-553 1990.
– reference: 4) 志村俊晴, 長尾寿, 他: 上顎骨中に埋伏歯を伴う血友病性血液嚢胞の手術治験例について. 交通医学33: 103-109 1979.
– reference: 5) Marquets, J. L., Vinageras, E., et al.: Hemophilic pseudotumor of the inferior maxilla. Oral Surg Oral Med Oral Pathol 53: 347-350 1982.
– reference: 8) Brook, A. H., Bedi, R., et al.: Hemophilic pseudotumor of the mandible: report of a case in a one-year-old child. Br J Oral Maxillofac Surg 23: 47-52 1985.
– reference: 14) Abell, J. M. and Bailey R. W.: Hemophilic pseudotumor: two cases occuring in sublings. Arch Surg 81: 569-581 1960.
– reference: 3) Mulkey, T. F.: Hemophilic pseudotumor of the mandible. J Oral Surg 35: 561-568 1977.
– reference: 9) 工藤正文, 横山浩之, 他: 血友病性偽腫瘍の1例. 八戸市民病院医誌12: 16-19 1989.
– reference: 6) Martinez-Lage, J. L., Sanchez, H., et al.: A pseudotumor of the mandible in a haemophiliac patient. J Maxillofac Surg 11: 171-173 1983.
– reference: 12) Zheng, K. and Zheng, P.: Hemophilic pseudotumor involving maxilla and tibia. Chin Med J 110: 233-235 1997.
– reference: 13) 川中正雄, 柳澤高道, 他: 幼児に生じた下顎骨血友病性偽腫瘍の1例. 口科誌46: 414-418 1997.
– reference: 7) Correra, A., Buckley, J., et al.: Radiotherapy of a pseudotumor in a hemophiliac with factor Mil inhibitor. Am J Pediatr Hematol Oncol 6: 325-327 1984.
– reference: 11) Sousa, S. M., Piratininga, J. L., et al.: Hemophilic pseudotumor of the jaws: report of a cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 79: 216-219 1995.
– reference: 2) Stoneman, D. W. and Beierl, C. D.: Pseudotumor of hemophilia in mandible. Oral Surg Oral Med Oral Pathol 40: 811-815 1975.
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Snippet A hemophilic pseudotumor was found in the mandible a of a 13-year-old boy with a known history of moderate hemophilia A. Intraorally, the alveolus was enlarged...
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SubjectTerms hemophilia A
hemophilic pseudotumor
mandible
Title A case of mandibular hemophilic pseudotumor
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