Orofacial granulomatosis を初発したクローン病の１例
Crohn's disease (CD) is a progressive, idiopathic granulomatous inflammatory disease that can affect any location of the gastrointestinal tract from the mouth to the anus. Orofacial granulomatosis (OFG) has been reported to occur as a manifestation of CD in young individuals. This report presen...
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| Published in | 日本口腔外科学会雑誌 Vol. 71; no. 2; pp. 63 - 69 |
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| Main Authors | , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
公益社団法人 日本口腔外科学会
20.02.2025
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0021-5163 2186-1579 |
| DOI | 10.5794/jjoms.71.63 |
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| Abstract | Crohn's disease (CD) is a progressive, idiopathic granulomatous inflammatory disease that can affect any location of the gastrointestinal tract from the mouth to the anus. Orofacial granulomatosis (OFG) has been reported to occur as a manifestation of CD in young individuals. This report presents the case of an 11-year-old girl who was diagnosed with CD two years after the onset of OFG. Oral examination revealed diffuse enlargement of both lips and hyperplastic gingival enlargement in the anterior regions. These observations were consistent with OFG. Histopathological examination of a gingival biopsy reported the presence of noncaseating, epithelioid-type granulomas. Two years later, the patient was hospitalized for hematochezia. The diagnosis of CD was made by her gastroenterologist. After initiating biologic therapy, both intestinal and OFG symptoms went into remission. OFG may occur for a variety of reasons. Particular attention should be paid to pediatric OFG, which may precede gastrointestinal lesions. Clinicians should consider CD in the differential diagnosis. |
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| AbstractList | Crohn's disease (CD) is a progressive, idiopathic granulomatous inflammatory disease that can affect any location of the gastrointestinal tract from the mouth to the anus. Orofacial granulomatosis (OFG) has been reported to occur as a manifestation of CD in young individuals. This report presents the case of an 11-year-old girl who was diagnosed with CD two years after the onset of OFG. Oral examination revealed diffuse enlargement of both lips and hyperplastic gingival enlargement in the anterior regions. These observations were consistent with OFG. Histopathological examination of a gingival biopsy reported the presence of noncaseating, epithelioid-type granulomas. Two years later, the patient was hospitalized for hematochezia. The diagnosis of CD was made by her gastroenterologist. After initiating biologic therapy, both intestinal and OFG symptoms went into remission. OFG may occur for a variety of reasons. Particular attention should be paid to pediatric OFG, which may precede gastrointestinal lesions. Clinicians should consider CD in the differential diagnosis. |
| Author | 末光, 正昌 田中, 茂男 久山, 佳代 大島, 麻耶 |
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| References | 18) Muhvić-Urek M, Tomac-Stojmenovic M, at al : Oral pathology in inflammatory bowel disease. World J Gastroenterol 22: 5655-5667, 2016. 9) Chen KL, Diiorio DA, et al : Pediatric patients with orofacial granulomatosis likely to subsequently develop intestinal Crohn's disease: Brief Report. Pediatr Dermatol 37: 1162-1164, 2020. 11) Phillips F, Verstockt B, et al : Orofacial granulomatosis associated with Crohn's disease: a multicentre case series. J Crohns Colitis 16: 430-435, 2022. 13) 鈴木茉莉恵，猿田祐輔，他：【肉芽腫性炎症】臨床例肉芽腫性口唇炎を契機に診断されたCrohn 病．皮病診療 37: 767-770, 2015. 12) Koga M, Koga K, at al : Favorable response to composite treatment including infliximab in a case of granulomatous cheilitis with Crohn's disease. J Dermatol 42: 651-652, 2015. 2) Rees TD : Orofacial granulomatosis and related conditions. Periodontol 2000 21: 145-57, 1999. 3) Endo H and Rees TD : Cinnamon products as a possible etiologic factor in orofacial granulomatosis. Med Oral Patol Oral Cir Bucal 12: 440-444, 2007. 15) 石井広太郎，室屋龍祐，他：口腔症状を主症状としたクローン病の１例．日口外誌 66: 627-632, 2020. 19) Hullah EA and Escudier MP : The mouth in inflammatory bowel disease and aspects of orofacial granulomatosis. Periodontol 2000 80: 61-76, 2019. 16) 下間祐輝，尾古俊哉，他：口腔症状を契機とし，Crohn病と診断した１例．日口腔診断会誌 37: 19-24, 2024. 5) 河 瑠珠，神部芳則，他：Orofacial granulomatosisの２例．日口腔粘膜会誌 15: 81-87, 2009. 6) 山下雅子，神部芳則，他：歯肉に生じたOrofacial granulomatosisの１例．日口腔内会誌 23: 34-39, 2017. 7) Kimura T, Sakata K, at al : Orofacial granulomatosis among pediatric patients well controlled by corticosteroid treatment: A rare case series. Case Reports in Pediatrics 2024: 1-6, 2024. 14) 角田和之，佐藤英和，他：歯肉腫脹を初発症状としたクローン病の１例．日口腔内会誌 23: 84-89, 2017. 20) Neville JJ, Beattie RM, at al : The diagnostic utility of lip biopsy in paediatric Crohn's disease: A 10-year single-centre retrospective study. J Pediatr Surg 59: 258-260, 2024. 17) 平山賀士子，狩野葉子，他：肉芽腫性口唇炎が先行したCrohn病の２例．皮膚臨床 39: 165-168, 1997. 1) 令和５年度改訂版 潰瘍性大腸炎・クローン病 診断基準・治療指針．厚生労働科学研究費補助金難治性疾患政策研究事業「難治性炎症性腸管障害に関する調査研究」(久松班) 令和５年度分担研究報告書．Aveilable at: http://www.ibdjapan.org/pdf/doc15.pdf. Accessed May 19, 2024. 8) Lazzerini M, Bramuzzo M, et al : Association between orofacial granulomatosis and Crohn's disease in children: systematic review. World J Gastroenterol 20: 7497-7504, 2014. 4) Jinbu Y, Tsukinoki K, at al : Orofacial Granulomatosis: Report of a case. Oral Medicine & Pathology 7: 33-37, 2002. 10) Gavioli CFB, Florezi GP, at al : Orofacial Granulomatosis and Crohn Disease: Coincidence or Pattern? A Systematic Review. Dermatology 237: 635-640, 2021. |
| References_xml | – reference: 8) Lazzerini M, Bramuzzo M, et al : Association between orofacial granulomatosis and Crohn's disease in children: systematic review. World J Gastroenterol 20: 7497-7504, 2014. – reference: 18) Muhvić-Urek M, Tomac-Stojmenovic M, at al : Oral pathology in inflammatory bowel disease. World J Gastroenterol 22: 5655-5667, 2016. – reference: 3) Endo H and Rees TD : Cinnamon products as a possible etiologic factor in orofacial granulomatosis. Med Oral Patol Oral Cir Bucal 12: 440-444, 2007. – reference: 12) Koga M, Koga K, at al : Favorable response to composite treatment including infliximab in a case of granulomatous cheilitis with Crohn's disease. J Dermatol 42: 651-652, 2015. – reference: 6) 山下雅子，神部芳則，他：歯肉に生じたOrofacial granulomatosisの１例．日口腔内会誌 23: 34-39, 2017. – reference: 10) Gavioli CFB, Florezi GP, at al : Orofacial Granulomatosis and Crohn Disease: Coincidence or Pattern? A Systematic Review. Dermatology 237: 635-640, 2021. – reference: 5) 河 瑠珠，神部芳則，他：Orofacial granulomatosisの２例．日口腔粘膜会誌 15: 81-87, 2009. – reference: 2) Rees TD : Orofacial granulomatosis and related conditions. Periodontol 2000 21: 145-57, 1999. – reference: 4) Jinbu Y, Tsukinoki K, at al : Orofacial Granulomatosis: Report of a case. Oral Medicine & Pathology 7: 33-37, 2002. – reference: 7) Kimura T, Sakata K, at al : Orofacial granulomatosis among pediatric patients well controlled by corticosteroid treatment: A rare case series. Case Reports in Pediatrics 2024: 1-6, 2024. – reference: 14) 角田和之，佐藤英和，他：歯肉腫脹を初発症状としたクローン病の１例．日口腔内会誌 23: 84-89, 2017. – reference: 19) Hullah EA and Escudier MP : The mouth in inflammatory bowel disease and aspects of orofacial granulomatosis. Periodontol 2000 80: 61-76, 2019. – reference: 13) 鈴木茉莉恵，猿田祐輔，他：【肉芽腫性炎症】臨床例肉芽腫性口唇炎を契機に診断されたCrohn 病．皮病診療 37: 767-770, 2015. – reference: 15) 石井広太郎，室屋龍祐，他：口腔症状を主症状としたクローン病の１例．日口外誌 66: 627-632, 2020. – reference: 1) 令和５年度改訂版 潰瘍性大腸炎・クローン病 診断基準・治療指針．厚生労働科学研究費補助金難治性疾患政策研究事業「難治性炎症性腸管障害に関する調査研究」(久松班) 令和５年度分担研究報告書．Aveilable at: http://www.ibdjapan.org/pdf/doc15.pdf. Accessed May 19, 2024. – reference: 9) Chen KL, Diiorio DA, et al : Pediatric patients with orofacial granulomatosis likely to subsequently develop intestinal Crohn's disease: Brief Report. Pediatr Dermatol 37: 1162-1164, 2020. – reference: 17) 平山賀士子，狩野葉子，他：肉芽腫性口唇炎が先行したCrohn病の２例．皮膚臨床 39: 165-168, 1997. – reference: 20) Neville JJ, Beattie RM, at al : The diagnostic utility of lip biopsy in paediatric Crohn's disease: A 10-year single-centre retrospective study. J Pediatr Surg 59: 258-260, 2024. – reference: 11) Phillips F, Verstockt B, et al : Orofacial granulomatosis associated with Crohn's disease: a multicentre case series. J Crohns Colitis 16: 430-435, 2022. – reference: 16) 下間祐輝，尾古俊哉，他：口腔症状を契機とし，Crohn病と診断した１例．日口腔診断会誌 37: 19-24, 2024. |
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| SubjectTerms | クローン病 口腔顔面肉芽腫症 非乾酪性類上皮細胞肉芽腫 |
| Title | Orofacial granulomatosis を初発したクローン病の１例 |
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