遺伝性血管性浮腫の小児に対して下顎の含歯性嚢胞摘出術を行った1例

We report the case of an 8-years-old child with hereditary angioedema who required oral surgery. Hereditary angioedema (HAE) is a rare autosomal dominant hereditary disease caused by a functional disorder of C1 inhibitor (C1-INH) or a low level of C1- INH protein. HAE is characterized by repeated lo...

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Published in日本口腔外科学会雑誌 Vol. 59; no. 3; pp. 192 - 196
Main Authors 山城, 正司, 鵜澤, 成一, 山口, 聰, 原田, 清, 園田, 格
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.03.2013
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ISSN0021-5163
2186-1579
DOI10.5794/jjoms.59.192

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Abstract We report the case of an 8-years-old child with hereditary angioedema who required oral surgery. Hereditary angioedema (HAE) is a rare autosomal dominant hereditary disease caused by a functional disorder of C1 inhibitor (C1-INH) or a low level of C1- INH protein. HAE is characterized by repeated localized edema of submucocutaneous tissues. Attacks are caused by trauma, drugs, infection, or mental stress. Dental treatment and oral surgery in patients with HAE can trigger fatal laryngeal edema. To prevent angioedema attacks, we injected C1-INH before surgery in a child with HAE. She underwent tooth extraction and cystectomy of the left side of the mandible under general anestethia. No HAE-related complications occurred during or after surgery, and perioperative management went well.
AbstractList We report the case of an 8-years-old child with hereditary angioedema who required oral surgery. Hereditary angioedema (HAE) is a rare autosomal dominant hereditary disease caused by a functional disorder of C1 inhibitor (C1-INH) or a low level of C1- INH protein. HAE is characterized by repeated localized edema of submucocutaneous tissues. Attacks are caused by trauma, drugs, infection, or mental stress. Dental treatment and oral surgery in patients with HAE can trigger fatal laryngeal edema. To prevent angioedema attacks, we injected C1-INH before surgery in a child with HAE. She underwent tooth extraction and cystectomy of the left side of the mandible under general anestethia. No HAE-related complications occurred during or after surgery, and perioperative management went well.
Author 園田, 格
山城, 正司
鵜澤, 成一
原田, 清
山口, 聰
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References 7) 大澤 勲, 大井洋之, 他:遺伝性血管性浮腫(HAE)を見落としていないだろうか?-全国の救命救急センターへのアンケート調査で判明した臨床現場での低い疾患認知度-.MEDICAL TORCH 5: 1-4, 2009.
27) Bork K, Hardt J, et al : Risk of laryngeal edema and facial swellings after tooth extraction in patients with hereditary angioedema with and without prophylaxis with C1 inhibitor concentrate: a retrospective study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 112: 58-64, 2011.
17) 大渡凡人,海野雅浩,他:遺伝性血管神経性浮腫(Hereditary angioneurotic edema)を有する高齢歯科患者の全身管理経験.日歯麻誌 23: 412-420, 1995.
13) Chue PWY : Acute angioneurotic edema of the lips and tongue due to emotional stress. Oral Surg 41: 734-738, 1976.
10) Carugati A, Pappalardo E, et al : C1-inhibitor deficiency and angioedema. Mol Immunol 38: 161-173, 2001.
5) 補体研究会:HAEガイドライン2010 Available at :http://square.umin.ac.jp/compl/HAE/HAEGuideline2010.pdf Accesssed October 31, 2011.
2) Gadek JE, Hosea SW, et al : Response of variant hereditary angioedema phenotypes to danazol therapy. J Clin Invest 64: 280-286, 1979.
11) Han ED, MacFarlane RC, et al : Increased vascular permeanility in C1 inhibitor-deficient mice mediated by the bradykinin type2 receptor. J Clin Invest 109: 1057-1063, 2002.
3) Michael M, Frank MM, et al : Hereditary angioedema: the clinical syndrome and its managrment. Ann Intern Med 84: 580-593, 1976.
19) Maeda S, Miyawaki T, et al : Management of oral surgery in patients with hereditary or acquired angioedemas: review and case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 96: 540-543, 2003.
8) Osler W : Hereditary angio-neurotic edema. Am J med Sci 95: 362-366, 1888.
9) Binkley KE and Davis III A : Clinical, biochemical, and genetic characterization of a novel estrogendependent inherited form of angioedema. J Allergy Clin Immunol 106: 546-550, 2000.
4) Jaffe CJ, Atkinson JP, et al : Hereditary angioedema: The use of fresh frozen plasma for prophylaxis in patients undergoing oral surgery. J Allergy Clin Immunol 55: 386-393, 1975.
6) Visentin DE and Karsh J : C1-elastase inhibitor transfusions in patients with hereditary angioedema. Ann Allegy Asthma Immunol 80: 457-461, 1998.
20) 岩井俊憲,筑丸 寛,他:遺伝性血管神経性浮腫の1例-周術期管理を中心に-.日口外誌 51: 260-263, 2005.
26) 大澤 勲,長町誠爾,他:遺伝性血管性浮腫(hereditary angioedema; HAE)-疾患概要と疾患認知度全国調査-.Hereditary angioedema recoginitionsurvey in Japan. Pharma Medica 29: 109-118, 2011.
21) 雨宮 肇,懸 秀栄,他:遺伝性血管神経性浮腫(HAE)を有する患者の歯科治療経験.日歯麻誌 34:164-168, 2006.
22) 渡邉賀子,加藤文度,他:遺伝性血管性浮腫患者の抜歯経験.日口外誌 53: 38-41, 2007.
12) Lawrence CP : Hereditary angioedema: Diagnosis and management- a perspective for the dermatologist. J Am Acad Dermatol 65: 843-850, 2011.
25) Moraes RS, Vieira AS, et al : Management of a dentigerous cyst in a child with hereditary angioedema. J Dent Child (Chic). Jan-Apr 77: 59-63, 2010.
16) 高橋徳昭, 関戸幹夫, 他:遺伝性血管神経性浮腫の1例.口科誌 33: 111-118, 1984.
14) Moore GP, Hurley WT, et al : Hereditary angioedema. Ann Emerg Med 17: 1082-1086, 1988.
15) Bork K and Barnstedt SE : Laryngeal edema and death from asphyxiation after tooth extraction in four patients with hereditary angioedema. J Am Dent Assoc 134 : 1088-1094, 2003.
23) 中野旬之,三木哲英,他:遺伝性血管性浮腫患者の抜歯経験.日口外誌 55: 136-139, 2009.
24) 濱田正和,奥長秀介,他:C1インヒビター投与下に嚢胞摘出術を行った遺伝性血管浮腫の1例.日口外誌 57: 74-77, 2011.
1) Rosen FS, Alper CA, et al : Genetically determined heterogeneity of C1 esterase inhibitor in patients with hereditary angioneurotic edema. J Clin Invest 50: 2143-2149, 1971.
18) 坂田康彰,西川久美子,他:遺伝性血管神経性浮腫を有する患者の歯科治療経験.日口外誌 49: 470-473, 2003.
References_xml – reference: 7) 大澤 勲, 大井洋之, 他:遺伝性血管性浮腫(HAE)を見落としていないだろうか?-全国の救命救急センターへのアンケート調査で判明した臨床現場での低い疾患認知度-.MEDICAL TORCH 5: 1-4, 2009.
– reference: 19) Maeda S, Miyawaki T, et al : Management of oral surgery in patients with hereditary or acquired angioedemas: review and case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 96: 540-543, 2003.
– reference: 27) Bork K, Hardt J, et al : Risk of laryngeal edema and facial swellings after tooth extraction in patients with hereditary angioedema with and without prophylaxis with C1 inhibitor concentrate: a retrospective study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 112: 58-64, 2011.
– reference: 9) Binkley KE and Davis III A : Clinical, biochemical, and genetic characterization of a novel estrogendependent inherited form of angioedema. J Allergy Clin Immunol 106: 546-550, 2000.
– reference: 1) Rosen FS, Alper CA, et al : Genetically determined heterogeneity of C1 esterase inhibitor in patients with hereditary angioneurotic edema. J Clin Invest 50: 2143-2149, 1971.
– reference: 3) Michael M, Frank MM, et al : Hereditary angioedema: the clinical syndrome and its managrment. Ann Intern Med 84: 580-593, 1976.
– reference: 13) Chue PWY : Acute angioneurotic edema of the lips and tongue due to emotional stress. Oral Surg 41: 734-738, 1976.
– reference: 4) Jaffe CJ, Atkinson JP, et al : Hereditary angioedema: The use of fresh frozen plasma for prophylaxis in patients undergoing oral surgery. J Allergy Clin Immunol 55: 386-393, 1975.
– reference: 15) Bork K and Barnstedt SE : Laryngeal edema and death from asphyxiation after tooth extraction in four patients with hereditary angioedema. J Am Dent Assoc 134 : 1088-1094, 2003.
– reference: 17) 大渡凡人,海野雅浩,他:遺伝性血管神経性浮腫(Hereditary angioneurotic edema)を有する高齢歯科患者の全身管理経験.日歯麻誌 23: 412-420, 1995.
– reference: 14) Moore GP, Hurley WT, et al : Hereditary angioedema. Ann Emerg Med 17: 1082-1086, 1988.
– reference: 11) Han ED, MacFarlane RC, et al : Increased vascular permeanility in C1 inhibitor-deficient mice mediated by the bradykinin type2 receptor. J Clin Invest 109: 1057-1063, 2002.
– reference: 24) 濱田正和,奥長秀介,他:C1インヒビター投与下に嚢胞摘出術を行った遺伝性血管浮腫の1例.日口外誌 57: 74-77, 2011.
– reference: 6) Visentin DE and Karsh J : C1-elastase inhibitor transfusions in patients with hereditary angioedema. Ann Allegy Asthma Immunol 80: 457-461, 1998.
– reference: 20) 岩井俊憲,筑丸 寛,他:遺伝性血管神経性浮腫の1例-周術期管理を中心に-.日口外誌 51: 260-263, 2005.
– reference: 5) 補体研究会:HAEガイドライン2010 Available at :http://square.umin.ac.jp/compl/HAE/HAEGuideline2010.pdf Accesssed October 31, 2011.
– reference: 12) Lawrence CP : Hereditary angioedema: Diagnosis and management- a perspective for the dermatologist. J Am Acad Dermatol 65: 843-850, 2011.
– reference: 16) 高橋徳昭, 関戸幹夫, 他:遺伝性血管神経性浮腫の1例.口科誌 33: 111-118, 1984.
– reference: 2) Gadek JE, Hosea SW, et al : Response of variant hereditary angioedema phenotypes to danazol therapy. J Clin Invest 64: 280-286, 1979.
– reference: 21) 雨宮 肇,懸 秀栄,他:遺伝性血管神経性浮腫(HAE)を有する患者の歯科治療経験.日歯麻誌 34:164-168, 2006.
– reference: 10) Carugati A, Pappalardo E, et al : C1-inhibitor deficiency and angioedema. Mol Immunol 38: 161-173, 2001.
– reference: 23) 中野旬之,三木哲英,他:遺伝性血管性浮腫患者の抜歯経験.日口外誌 55: 136-139, 2009.
– reference: 18) 坂田康彰,西川久美子,他:遺伝性血管神経性浮腫を有する患者の歯科治療経験.日口外誌 49: 470-473, 2003.
– reference: 26) 大澤 勲,長町誠爾,他:遺伝性血管性浮腫(hereditary angioedema; HAE)-疾患概要と疾患認知度全国調査-.Hereditary angioedema recoginitionsurvey in Japan. Pharma Medica 29: 109-118, 2011.
– reference: 25) Moraes RS, Vieira AS, et al : Management of a dentigerous cyst in a child with hereditary angioedema. J Dent Child (Chic). Jan-Apr 77: 59-63, 2010.
– reference: 22) 渡邉賀子,加藤文度,他:遺伝性血管性浮腫患者の抜歯経験.日口外誌 53: 38-41, 2007.
– reference: 8) Osler W : Hereditary angio-neurotic edema. Am J med Sci 95: 362-366, 1888.
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Snippet We report the case of an 8-years-old child with hereditary angioedema who required oral surgery. Hereditary angioedema (HAE) is a rare autosomal dominant...
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喉頭浮腫
遺伝性血管性浮腫
Title 遺伝性血管性浮腫の小児に対して下顎の含歯性嚢胞摘出術を行った1例
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