重症型血友病Bの幼児に生じた上顎骨血友病性偽腫瘍の1例

Hemophilic pseudotumor causes hematoma mainly in the joints and muscles of hemophilia patients. It rarely occurs in the jawbone. We describe our experience with a hemophilic pseudotumor in the maxilla of an infant aged 1 year, 4 months. The patient was being followed-up for severe hemophilia B in ou...

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Published in日本口腔外科学会雑誌 Vol. 66; no. 11; pp. 572 - 576
Main Authors 川尻, 秀一, 加藤, 広禄, 小林, 一彦, 小林, 久乃, 北原, 寛子, 宮澤, 広樹
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.11.2020
Subjects
幼児
血友病B
血友病性偽腫瘍
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ISSN0021-5163
2186-1579
DOI10.5794/jjoms.66.572

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Abstract Hemophilic pseudotumor causes hematoma mainly in the joints and muscles of hemophilia patients. It rarely occurs in the jawbone. We describe our experience with a hemophilic pseudotumor in the maxilla of an infant aged 1 year, 4 months. The patient was being followed-up for severe hemophilia B in our hospital’s department of pediatrics. At the initial visit, swelling of the buccal gingiva of the maxilla second deciduous molar was observed, and CT images showed a tumor in the maxillary left molar region. After the examination, repeated bleeding from the gingival sulcus due to mobility of the maxillary second deciduous molar occurred. Therefore, following the guidelines, we administered a blood coagulation factor IX preparation and extracted the maxillary second deciduous molar under local anesthesia. About 1 month later, we removed the maxillary lesion with the patient under general anesthesia. The lesion was covered with a thin capsule and filled with blood, and the crown of the maxillary first molar was contained within the lesion. The histopathological findings showed internal blood retention and reactive granulation augmentation. The lesion was diagnosed as a hemophilic pseudotumor. The postoperative course has been good with no recurrence of the lesion for 2 years.
AbstractList Hemophilic pseudotumor causes hematoma mainly in the joints and muscles of hemophilia patients. It rarely occurs in the jawbone. We describe our experience with a hemophilic pseudotumor in the maxilla of an infant aged 1 year, 4 months. The patient was being followed-up for severe hemophilia B in our hospital’s department of pediatrics. At the initial visit, swelling of the buccal gingiva of the maxilla second deciduous molar was observed, and CT images showed a tumor in the maxillary left molar region. After the examination, repeated bleeding from the gingival sulcus due to mobility of the maxillary second deciduous molar occurred. Therefore, following the guidelines, we administered a blood coagulation factor IX preparation and extracted the maxillary second deciduous molar under local anesthesia. About 1 month later, we removed the maxillary lesion with the patient under general anesthesia. The lesion was covered with a thin capsule and filled with blood, and the crown of the maxillary first molar was contained within the lesion. The histopathological findings showed internal blood retention and reactive granulation augmentation. The lesion was diagnosed as a hemophilic pseudotumor. The postoperative course has been good with no recurrence of the lesion for 2 years.
Author 北原, 寛子
加藤, 広禄
宮澤, 広樹
川尻, 秀一
小林, 一彦
小林, 久乃
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References 3) 天野景裕， 大平勝美， 他：厚生労働省委託事業 平成28年度 (2016年度) 血液凝固異常症全国調査報告書．API-Net, 公益財団法人エイズ予防財団：2017. Available at:http://api-net.jfap.or.jp/library/alliedEnt/02 Accessed April 16, 2019.
4) Starker L : Knochenusur durch ein hamophiles, subperiostales Hamatom. Mitt Grenzgeb Med Chir 31: 381, 1918.
18) Stonema DW and Beierl CD : Pseudotumor of hemophilia in the mandible: Report of a case. Oral Surg Oral Med Oral Pathol 40: 811-815, 1975.
6) Lee DW, Jee YJ, et al : Changes in a hemophilic pseudotumor of the mandible that developed over a 20-year period. J oral Maxillofac Surg 67: 1516-1520, 2009.
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.
17) 平井雄三， 竹信俊彦， 他：上顎骨に発生した 血友病性偽腫瘍の1例．日口外誌 64: 92-97, 2018.
12) 工藤正文， 横山浩之， 他：血友病性偽腫瘍の1例．八戸病医誌 12: 16-19, 1989.
14) 河原　康, 堀田文雄, 他: 下顎骨にみられた血友病性偽腫瘍の1例. 日口外誌 46: 105-107, 2000.
7) Cox DP, SolarA, et al : Pseudotumor of the mandible as frst presentation of hemophilia in a 2-year-old male: a case report and review of jaw pseudotumors of hemophilia. Head Neck Pathol 5: 226-232, 2011.
15) Yoshitake Y, Nakayama H, et al : Haemophilic pseudotumour of the mandible in a 5-year-old patient. Int J Oral Maxillofac Surg 40: 106-123, 2011.
8) Srinivasan K, Gadodia A, et al : Magnetic resonance imaging of mandibular hemophilic pseudotumor associated with factor Ⅸ defciency: report of case with review of literature. J Oral Maxillofac Surg 69: 1683-1690, 2011.
11) 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囊胞の手術治験例について．交通医33: 103-109, 1979.
16) Otsuka H, Ozeki M, et al : Complete bone regeneration in hemophilic pseudotumor of the mandible. Pediatr Int 5: 406-408, 2016.
2) 藤井輝久， 天野景裕， 他：インヒビターのない血友病患者に対する止血治療ガイドライ2013年改訂版．日血栓止血会誌 24: 619-639, 2013.
1) 竹谷英之：血友病症例の手術と止血管理．日血栓止血会誌 18: 580-583, 2007.
9) Kwon AY, Huh KH, et al : haemophilic pseudotumor in two parts of the maxilla: case report. Dentomaxillofac Radiol 45: 20150440, 2016.
10) Probodh KC, Shakil AN, et al : Hemophilic pseudotumor of the mandible in a patient with hemophilia B. Oral Maxillofac Surg 21: 467-469, 2017.
5) Lazarovits P and Griem ML : Radiotherapy of hemophiliac pseudotumors. Radiology 91: 1026-1027, 1968.
References_xml – reference: 2) 藤井輝久， 天野景裕， 他：インヒビターのない血友病患者に対する止血治療ガイドライ2013年改訂版．日血栓止血会誌 24: 619-639, 2013.
– reference: 3) 天野景裕， 大平勝美， 他：厚生労働省委託事業 平成28年度 (2016年度) 血液凝固異常症全国調査報告書．API-Net, 公益財団法人エイズ予防財団：2017. Available at:http://api-net.jfap.or.jp/library/alliedEnt/02 Accessed April 16, 2019.
– reference: 4) Starker L : Knochenusur durch ein hamophiles, subperiostales Hamatom. Mitt Grenzgeb Med Chir 31: 381, 1918.
– reference: 6) Lee DW, Jee YJ, et al : Changes in a hemophilic pseudotumor of the mandible that developed over a 20-year period. J oral Maxillofac Surg 67: 1516-1520, 2009.
– reference: 8) Srinivasan K, Gadodia A, et al : Magnetic resonance imaging of mandibular hemophilic pseudotumor associated with factor Ⅸ defciency: report of case with review of literature. J Oral Maxillofac Surg 69: 1683-1690, 2011.
– reference: 7) Cox DP, SolarA, et al : Pseudotumor of the mandible as frst presentation of hemophilia in a 2-year-old male: a case report and review of jaw pseudotumors of hemophilia. Head Neck Pathol 5: 226-232, 2011.
– 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: 5) Lazarovits P and Griem ML : Radiotherapy of hemophiliac pseudotumors. Radiology 91: 1026-1027, 1968.
– reference: 10) Probodh KC, Shakil AN, et al : Hemophilic pseudotumor of the mandible in a patient with hemophilia B. Oral Maxillofac Surg 21: 467-469, 2017.
– reference: 16) Otsuka H, Ozeki M, et al : Complete bone regeneration in hemophilic pseudotumor of the mandible. Pediatr Int 5: 406-408, 2016.
– reference: 14) 河原　康, 堀田文雄, 他: 下顎骨にみられた血友病性偽腫瘍の1例. 日口外誌 46: 105-107, 2000.
– reference: 17) 平井雄三， 竹信俊彦， 他：上顎骨に発生した 血友病性偽腫瘍の1例．日口外誌 64: 92-97, 2018.
– reference: 1) 竹谷英之：血友病症例の手術と止血管理．日血栓止血会誌 18: 580-583, 2007.
– reference: 9) Kwon AY, Huh KH, et al : haemophilic pseudotumor in two parts of the maxilla: case report. Dentomaxillofac Radiol 45: 20150440, 2016.
– reference: 11) 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囊胞の手術治験例について．交通医33: 103-109, 1979.
– reference: 15) Yoshitake Y, Nakayama H, et al : Haemophilic pseudotumour of the mandible in a 5-year-old patient. Int J Oral Maxillofac Surg 40: 106-123, 2011.
– reference: 12) 工藤正文， 横山浩之， 他：血友病性偽腫瘍の1例．八戸病医誌 12: 16-19, 1989.
– reference: 18) Stonema DW and Beierl CD : Pseudotumor of hemophilia in the mandible: Report of a case. Oral Surg Oral Med Oral Pathol 40: 811-815, 1975.
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SubjectTerms 幼児
血友病B
血友病性偽腫瘍
Title 重症型血友病Bの幼児に生じた上顎骨血友病性偽腫瘍の1例
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