• Restorative Dentistry and Endodontics
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• 제47권3호
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• pp.26.1-26.10
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• 2022

Wegener's granulomatosis (WG) is a condition with immune-mediated pathogenesis that can present oral manifestations. This report describes the case of a patient diagnosed with WG 14 years previously, who was affected by persistent pain of non-odontogenic origin after successful endodontic treatment. A 39-year-old woman with WG was diagnosed with pulp necrosis and apical periodontitis of teeth #31, #32, and #41, after evaluation through a clinical examination and cone-beam computed tomography (CBCT). At the first appointment, these teeth were subjected to conventional endodontic treatment. At 6- and 12-month follow-up visits, the patient complained of persistent pain associated with the endodontically treated teeth (mainly in tooth #31), despite complete remission of the periapical lesions shown by radiographic and CBCT exams proving the effectiveness of the endodontic treatments, thus indicating a probable diagnostic of persistent pain of non-odontogenic nature. After the surgical procedure was performed to curette the lesion and section 3 mm of the apical third of tooth #31, the histopathological analysis suggested that the painful condition was likely associated with the patient's systemic condition. Based on clinical, radiographic, and histopathological findings, this unusual case report suggests that WG may be related to non-odontogenic persistent pain after successful endodontic treatments.

• Maxillofacial Plastic and Reconstructive Surgery
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• 제34권3호
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• pp.220-225
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• 2012

Wegener's granulomatosis (WG) is an autoimmune disease of unknown etiology characterized by the triad of necrotizing granulomatous lesion in the upper and lower respiratory tracts or both, disseminated vasculitis involving both small arteries and veins, and necrotizing glomerulonephritis. The most common oral lesions associated with WG are ulceration and strawberry gingivitis. A 47-years old man in medical care associated WG was consulted our Division of Oral and Maxillofacial Surgery for the chief complaint of toothaches. Pre-operative panorama showed the alveolar radiolucency and the loss of lamina dura regarding the left upper teeth. An oropharyngeal magnetic resonance imaging also revealed the increased bone marrow signal intensity on the left maxilla. Under the impression of maxillary osteonecrosis due to WG, maxillary saucerization with removal of involved teeth was performed. We obtained good results and report the first case of WG in Korea, with the review of literatures regarding oral and general systemic features.

• Tuberculosis and Respiratory Diseases
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• 제49권5호
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• pp.644-648
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• 2000

저자들은 고열을 동반한 피부 궤양과 폐의 다발성 공동을 보인 환자를 항결핵제로 치료하였으나 반응하지 않아 피부와 폐의 조직 검사를 시행한 결과 괴사성 육아종성 확인하고 c-ANCA 검사 Wegener 육아종증으로 확진하여 cyclophosphamide와 methylpredni-solone으로 치료하여 임상적 호전을 보인 예를 보고하는 바이다.

• Journal of Periodontal and Implant Science
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• 제45권6호
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• pp.247-251
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• 2015

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.

• 대한두경부종양학회지
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• 제39권1호
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• pp.27-31
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• 2023

Granulomatosis with polyangiitis (GPA), formerly known as Wegener's granulomatosis, is a rare immunological small vessel disease which usually affects respiratory tract and kidneys. However, salivary gland involvement in GPA is rare as a primary manifestation. We report a case of GPA with the primary presentation of submandibular gland involvement. A 48-year-old female patient presented submandibular swelling with a skin defect that lasted for 1 month. Although the biopsy result was chronic inflammation, the skin defect did not heal for a month. Further imaging study revealed multiple lung and renal masses. More clinical manifestations such as gingivitis, ischemic change of finger joint and nasal tip skin, and positive c-ANCA test was presented. Additional biopsy was made at the submandibular gland, lung, and finger skin. The patient was finally diagnosed with GPA and treated with steroid pulse therapy and cyclophosphamide. The patient showed improvement of prior clinical symptoms.

• Tuberculosis and Respiratory Diseases
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• 제45권6호
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• pp.1290-1297
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• 1998

본 증례는 평소 건강히 지내던 38세 여자에서 아토피나 천식과의 연관성이 없이 호흡기계 증상 및 폐침윤을 동반한 예로, 비특이적인 증상 및 방사선학적 소견으로 일반적인 폐침윤에 대한 치료에 반응하지 않고 병변이 좌폐상엽의 설상엽에 국한되어 있어 수술을 통해 확진 하였기에 문헌고찰과 함께 보고하는 바이다.

• Tuberculosis and Respiratory Diseases
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• 제53권4호
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• pp.463-469
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• 2002

저자들은 비폐색과 두통을 주소로 내원한 49세 남자에서 단백뇨 및 혈뇨와 병발된 다발성 폐결절을 발견하였고, c-ANCA와 조직검사를 통해 부비동, 폐, 신장을 침범한 Wegener씨 육아종증을 진단하고, cyclophosphamide와 스테로이드 병합투여로 증세가 호전되고, 흉부 방사선 사진에서도 호전된 소견을 보이는 환자를 경험하였기에 문헌고찰과 함께 보고하는 바이다.

• Korean Journal of Radiology
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• 제2권4호
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• pp.222-230
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• 2001

Central diabetes insipidus (DI) can be the outcome of a number of diseases that affect the hypothalamic-neurohypophyseal axis. The causes of the condition can be classified as traumatic, inflammatory, or neoplastic. Traumatic causes include postoperative sella or transection of the pituitary stalk, while infectious or inflammatory causes include meningitis, lymphocytic hypophysitis, and granulomatous inflammations such as sarcoidosis and Wegener's granulomatosis. Various neoplastic conditions such as germinoma, Langerhans cell histiocytosis, metastasis, leukemic infiltration, lymphoma, teratoma, pituitary adenoma, craniopharyngioma, Rathke cleft cyst, hypothalamic glioma, and meningioma are also causes of central DI. In affected patients, careful analysis of these MR imaging features and correlation with the clinical manifestations can allow a more specific diagnosis, which is essential for treatment.

• Journal of Yeungnam Medical Science
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• 제21권1호
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• pp.101-107
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• 2004

Diffuse alveolar hemorrhage is a rare but serious and frequently life-threatening complication of a variety of conditions. The first goal in the management of patients with diffuse alveolar hemorrhage is to achieve or preserve stability of the respiratory status. Subsequently, the differential diagnosis is aimed at the identification of a remediable cause of the alveolar hemorrhage. The most common causes of diffuse alveolar hemorrhage with glomerulonephritis are microscopic polyangiitis and Wegener's granulomatosis, followed by Goodpasture syndrome and systemic lupus erythematosus. Microscopic polyangiitis (MPA) is a distinct systemic small vessle vasculitis affecting small sized vessels with few or no immune deposits and with no granulomatosus inflammation. The disease may involve multiple organs such as kidney, lung, skin, joint, muscle, gastrointestinal tract, eye, and nervous system. MPA is strongly associated with antineutrophil cytoplasmic autoantibody (ANCA) that is a useful serological diagnostic marker for the most common form of necrotizing vasculitis. Our report concerns a case of microscopic polyangiitis with diffuse alveolar hemorrhage in a 54-year-old man. He was admitted to our hospital due to dyspnea upon exertion and recurrent hemoptysis. Laboratory findings showed hematuria, proteinuria and deterioration of renal function. In the chest CT scan, diffuse ground glass appearance was seen in both lower lungs. A lung biopsy revealed small vessel vasculitis with intraalveolar hemorrhage and showed a positive reaction to against perinuclear ANCA. The patient was treated with prednisolone and cyclophosphamide. Chest infiltration decreased and hemoptysis and hypoxia improved. He is still being followed up in our hospital with a low dose of prednisolone.

• Tuberculosis and Respiratory Diseases
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• 제56권2호
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• pp.203-209
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• 2004

MTX는 골육종이나 진행된 비호치킨씨 임파종과 같은 종양성 질환 뿐아니라 건선이나 류마치스 관절염, 유육종증, 웨그너씨 육아종증등의 양성질환에서도 널리 쓰여지는 약물이다. MTX에 의한 폐독성은 약제를 투여받은 환자들 중 0.3-7.5%정도에서 나타나는 것으로 알려져 있다. 급성 또는 만성의 경과로 나타나는 MTX 폐렴의 예가 기술되어 있지만 전형적으로는 MTX를 투여한지 수개월내에 증상이 나타나는 아급성의 임상경과를 취하는 경우가 많다. 조직병리학적으로는 NSIP의 형태로 나타나는 경우가 가장 흔한 것으로 되어 있으나 BOOP나 DAD로 나타나는 경우도 드물게 보고되고 있으며 그 중 육아종의 형성을 보이는 예는 34.7% 정도로 알려져 있다. 우리나라에서는 NSIP나 DAD로 나타난 MTX 폐장염의 예가 보고되어있다. 저자들은 과민성 폐장염의 형태로 발현한 MTX에 의한 폐독성의 증례를 경험하였기에 문헌고찰과 함께 보고하는 바이다.