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Synchronous Orofacial Granulomatosis in a Patient with Brain Cavernous Hemangioma
윤승주,이지영,김광훈,정종헌,이애영,이승호,홍종수 대한피부과학회 2019 Annals of Dermatology Vol.31 No.6
Orofacial granulomatosis is a rare granulomatous inflammatory disease, characterized by recurrent orofacial swelling. Infectious, genetic, and immunologic etiologies are suggested, but not fully understood. Herein, we report a case of synchronous orofacial granulomatosis with brain cavernous hemangioma in a 44-year-old female patient, which may be considered paraneoplastic syndrome.
Synchronous Orofacial Granulomatosis in a Patient with Brain Cavernous Hemangioma
( Seung Ju Yun ),( Ji Young Lee ),( Gwang Hoon Kim ),( Jong Heon Jeong ),( Ai-young Lee ),( Seung Ho Lee ),( Jong Soo Hong ) 대한피부과학회 2019 Annals of Dermatology Vol.31 No.6
Orofacial granulomatosis is a rare granulomatous inflammatory disease, characterized by recurrent orofacial swelling. Infectious, genetic, and immunologic etiologies are suggested, but not fully understood. Herein, we report a case of synchronous orofacial granulomatosis with brain cavernous hemangioma in a 44-year-old female patient, which may be considered paraneoplastic syndrome. (Ann Dermatol 31(6) 678∼680, 2019)
권보람 ( Bo Ram Kwon ),최민정 ( Min Jung Choi ),변지연 ( Ji Yeon Byun ),최유원 ( You Won Choi ),최혜영 ( Hae Young Choi ) 대한피부과학회 2020 대한피부과학회지 Vol.58 No.3
Orofacial granulomatosis (OFG) is a rare chronic granulomatous disease of oral mucosa and maxillofacial region that clinically presents recurrent or persistent orofacial swelling. OFG may exist as an integration of the spectrum of various disorders, including Melkersson-Rosenthal syndrome, granulomatous cheilitis, Crohn’s disease, sarcoidosis, infectious diseases and delayed hypersensitivity reaction to food additives, dental materials. Precise etiology is not yet to be elucidated. Herein, we report two rare cases of orofacial granulomatosis who presented similar clinical manifestation of lip swelling but was diagnosed as a sarcoidosis and as a granulomatous cheilitis. (Korean J Dermatol 2020;58(3):191∼195)
Sung, Iel-Yong,Kim, Young-Min,Cho, Yeong-Cheol,Son, Jang-Ho Korean Academy of Periodontology 2015 Journal of Periodontal & Implant Science Vol.45 No.6
Purpose: This report describes a case of granulomatosis with polyangiitis (GPA) in which the gingival manifestation was crucial in both making an early diagnosis and possibly in deciding the approach to treatment. Methods: A 57-year-old sailor presented to the Department of Dentistry at Ulsan University Hospital complaining of gingival swelling since approximately 2 months. He had orofacial granulomatous lesions and the specific gingival manifestation of strawberry gingivitis. Results: The diagnosis of GPA was made on the basis of clinical symptoms and signs, and confirmed by the presence of the anti-neutrophil cytoplasmic antibody and a positive biopsy. The patient was admitted to the hospital and subsequently placed on a disease-modifying therapy regimen that included methotrexate and prednisone. Conclusions: Identification of the gingival manifestation of the disease permitted an early diagnosis and prompt therapy in a disease in which time is a crucial factor. Because of its rapid progression and potentially fatal outcome, an early diagnosis of GPA is important. Therefore, dentists should be aware of the oral signs and symptoms of such systemic diseases.
Orofacial granulomatosis in a 44-year-old woman with cavernous hemangioma of brain
( Seung Ju Yun ),( Gwang Hoon Kim ),( Ji Young Lee ),( Jong Heon Jeong ),( Ai-young Lee ),( Seung Ho Lee ),( Jong Soo Hong ) 대한피부과학회 2018 대한피부과학회 학술발표대회집 Vol.70 No.1
A 44-year-old woman visited our clinic with repeated labial and perilabial swelling for months. She had been treated with oral antiviral agents, but not effective. Recently, she was diagnosed with a brain cavernous hemangioma. Histologic examination revealed exocytosis of some lymphocytes and dense perivascular, perifollicular lymphohistiocytic infiltration throughout the dermis. Triamcinolone intralesional injection, oral methylprednisolone and doxycycline was not effective. Seven months after the first visit, stereotactic radiosurgery was performed for cavernous hemangioma. We prescribed the same dose of methylprednisolone as before. Despite dosage was not increased, symptoms tended to improve. After 2 months, oral methylprednisolone was discontinued and intermittent triamcinolone intralesional injection was performed solely. Her skin condition is improving continuously.
성일용,김영민,조영철,손장호 대한치주과학회 2015 Journal of Periodontal & Implant Science Vol.45 No.6
Purpose: This report describes a case of granulomatosis with polyangiitis (GPA) in whichthe gingival manifestation was crucial in both making an early diagnosis and possibly indeciding the approach to treatment. Methods: A 57-year-old sailor presented to the Department of Dentistry at Ulsan UniversityHospital complaining of gingival swelling since approximately 2 months. He had orofacialgranulomatous lesions and the specific gingival manifestation of strawberry gingivitis. Results: The diagnosis of GPA was made on the basis of clinical symptoms and signs, andconfirmed by the presence of the anti-neutrophil cytoplasmic antibody and a positive biopsy. The patient was admitted to the hospital and subsequently placed on a disease-modifyingtherapy regimen that included methotrexate and prednisone. Conclusions: Identification of the gingival manifestation of the disease permitted an earlydiagnosis and prompt therapy in a disease in which time is a crucial factor. Because of itsrapid progression and potentially fatal outcome, an early diagnosis of GPA is important. Therefore, dentists should be aware of the oral signs and symptoms of such systemic diseases.
Three Cases of Recurrent Cheilitis Granulomatosa Treated with Variable Combination Therapy
( Seung Pil Ham ),( Hee Jae Park ),( Cheong Ha Woo ),( Mira Choi ),( Hai-jin Park ) 대한피부과학회 2019 大韓皮膚科學會誌 Vol.57 No.4
Cheilitis granulomatosa (CG) is a subset of orofacial granulomatosis (OFG) and considered to be a monosymptomatic form of Melkersson-Rosenthal syndrome (MRS), which is characterized by the triad of chronic lip swelling, facial paralysis, and fissured tongue. The labial swelling is thought to be associated with an orofacial swelling, which affects the chin, cheeks, and oral mucosa. Histologically, it is distinguished by noncaseating granulomas consisting of lymphohistiocytes and giant cells. Although systemic steroids with or without intralesional triamcinolone injections are the mainstay of treatment, and various agents have been proposed for this rare disease, no successful treatment modality has been reported in the literature yet. Herein, we present our experience with three different CG cases, which showed a varied level of positive response to the combination therapy. (Korean J Dermatol 2019;57(4):209∼212)
Photobiomodulation and Miescher's cheilitis granulomatosa: case report
Porrini, Massimo,Garagiola, Umberto,Rossi, Margherita,Bosotti, Moreno,Marino, Sonia,Gianni, Aldo Bruno,Runza, Letterio,Spadari, Francesco Korean Association of Maxillofacial Plastic and Re 2020 Maxillofacial Plastic Reconstructive Surgery Vol.42 No.-
Background: Miescher's cheilitis granulomatosa (MCG) is a rare chronic inflammatory disease and is known as the monosymptomatic clinical form of Melkersson-Rosenthal syndrome (MRS). It is characterised by swelling of one or both lips and more frequently affects the upper lip. Histopathological findings show the presence of numerous inflammatory infiltrates and granuloma formations. Pharmacological treatments and surgery have provided results that are positive yet insufficiently stable in the long term. The clinical case described is of a 68-year-old female patient with a diagnosis of MCG of the upper lip. Case presentation: The patient was diagnosed and treated at the Oral Medicine and Oral Pathology outpatient clinic of Maxillofacial and Odontostomatology Unit, Fondazione Cà Granda IRCCS Ospedale Maggiore Policlinico. The patient was recommended localised treatments of photobiomodulation (PBM) using a diode laser with a 635 nm and 980 nm dual-wavelength (λ) approach, a 600-micron fibre, and a handpiece with a 1-cm-diameter lens at 300 mW. Three treatments a week were administered for four weeks for a total of 12 treatment sessions (T<sub>1</sub>-T<sub>12</sub>). After that, the patient had a long follow-up period of about 2 years. The therapeutic results were clear from the initial stages of treatment. There was an immediate, gradual, and consistent reduction in labial swelling. A reduction in the size of the lip by about 35% at T<sub>10</sub>-T<sub>12</sub> was observed, returning the size and volume of the upper lip within the normal clinical range. The painful symptoms subsided after the seventh treatment (T<sub>7</sub>). The histopathological check at 3 months and the follow-up in particular confirmed the disease was in remission with satisfactorily stable treatment results. Moreover, the patient did not use any other treatments on the area from the early laser treatments through to the end of the follow-up period. Conclusions: Our experience describes a clinical case of MCG treated with PBM and effectively resolved with a reduction of the lip swelling. The real success of the treatment emerged over time, showing that the tissue healing was stable. In absence of any collateral phenomena, this confirms the effective and documented therapeutic potential of PBM for chronic inflammatory infiltrates.