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1/9. Granulomatous cheilitis and Crohn's disease.

    Granulomatous cheilitis is characterized by recurrent swelling of the labial tissues and granulomatous histology. Granulomatous cheilitis has been recognized as an early manifestation of Crohn's disease. It may follow, coincide with or precede the onset of Crohn's disease. The first case presented involved an extraintestinal manifestation of Crohn's disease, and the second case presented is of development of granulomatous cheilitis a year before the onset of symptomatic Crohn's disease. Although chronic granulomatous cheilitis is a very rare disorder, once it is diagnosed, the patient should be followed up carefully. These patients should be investigated for asymptomatic Crohn's disease either when the diagnosis of granulomatous cheilitis is confirmed or when gastrointestinal symptoms develop.
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keywords = labial
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2/9. melkersson-rosenthal syndrome caused by saprodontia: a case report.

    A 24-year-old female patient with melkersson-rosenthal syndrome (MRS) in association with saprodontia is reported. She presented with lower labial swelling and left facial edema. Histological examination of the involved oral mucosa showed a noncaseating epithelioid granuloma. Results from the laboratory and imaging examinations were normal or negative. Her orofacial swelling disappeared after treatment of the saprodontia of the left first molar.
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keywords = labial
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3/9. cheilitis granulomatosa of melkersson-rosenthal syndrome: treatment with intralesional corticosteroid injections.

    BACKGROUND: melkersson-rosenthal syndrome may manifest as the classical triad (orofacial edema, facial nerve palsy and stable lingua plicata) but monosymptomatic manifestations or combinations of typical symptoms are not infrequent. The available therapeutic options provide only limited success or temporary benefit. CASE REPORT: A 20-year-old man presented with a 7-month history of recurrent episodes of swelling of the upper lip without pain, burning or local pruritus. No causative factors, such as food, drugs or latex, or physical, chemical or emotional conditions could be identified. The patient had been treated with oral antihistamines and corticosteroids with no clinical improvement. physical examination showed firm edema without fovea, limited to the central area of the upper lip without epidermal changes or symptoms on palpation. The patient had a previous history of facial palsy 6 years previously and recurrent episodes of herpes simplex labialis. skin prick tests with inhalant aeroallergens, food, latex and anisakis allergens were negative. Laboratory investigation revealed normal complete blood count, erythrocyte sedimentation rate, thyroid hormones, biochemistry, complement components (C3, C4 and C1-esterase inhibitor) and CH50, rheumatoid factor, antinuclear antibodies, immune complexes, protein electrophoresis and immunoglobulins. thorax and paranasal sinus radiographs were clear. biopsy of the involved area of the lip showed edema with lymphocytic and plasma cell infiltration and mononuclear perivascular infiltrates without granulomas, suggesting initial granulomatous cheilitis. Because the patient showed lack of response and/or poor tolerance to prior treatments (deflazacort, clofazimine and metronidazole), intralesional triamcinolone injections were administered with satisfactory response from the first session. CONCLUSIONS: Response to available treatments for melkersson-rosenthal syndrome is highly variable. In the present case, intralesional triamcinolone injections were effective.
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keywords = labial
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4/9. Management of cheilitis granulomatosa.

    Peripheral facial nerve palsy, recurrent or persistent oral or facial swelling, and fissured tongue constitute a triad of symptoms known as Melkersson-Rosenthal syndrome. Granulomatous labial enlargement, known as cheilitis granulomatosa, is considered the single most important diagnostic feature of this syndrome. This lesion has been difficult to treat. This article describes a case of 8 months' duration of cheilitis granulomatosa of the lower lip, which was successfully managed with intralesional steroid injections.
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keywords = labial
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5/9. melkersson-rosenthal syndrome in india: experience with six cases.

    Six patients with melkersson-rosenthal syndrome (M-R syndrome) were presented. Their ages varied from 20 to 60 years; age at onset was between 15 and 50 years. In all 6 patients, swelling started first over the lower lip. Associated facial palsy, facial edema, and scrotal tongue were found in three, four and one patients, respectively. lip biopsies from 5 patients failed to show any granuloma. Intralesional corticosteroids produced mild regression of labial swelling in two patients after 6 injections. In one patient, near total reduction of labial swelling and facial edema was achieved after 5 months of clofazimine treatment. However, there was no improvement of facial palsy. To the best of our knowledge, this is the largest series of M-R syndrome reported from india.
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keywords = labial
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6/9. Granulomatous glossitis as an unusual manifestation of melkersson-rosenthal syndrome.

    The case of a 59-year-old man with granulomatous glossitis is presented. The patient complained of increasing swelling of his tongue for years and a recent onset of perioral edema. Because of sarcoid-like granulomas found in a first lingual biopsy specimen, the disease was initially misdiagnosed as a localized form of sarcoidosis. A second biopsy specimen from the tongue revealed lymphonodular-plasmocytic granuloma-like infiltrates, which, in connection with the labial swelling, led to the diagnosis of Schuermann's glossitis granulomatosa as a peculiar manifestation of melkersson-rosenthal syndrome. The surface of the tongue showed deep furrows characteristic of lingua plicata. However, no history or signs of facial or trigeminal nerve paralysis were present; thus the patient did not show the complete triad of symptoms typical of melkersson-rosenthal syndrome. After treatment with clofazimine the perioral and lingual swelling disappeared within two weeks. Recurrences have been suppressed with a reduced maintenance dosage of this agent.
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keywords = labial
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7/9. Orofacial lesions in Crohn's disease.

    We describe a female patient with Crohn's disease who presented with longstanding gingival swelling. She went into remission after surgery, but had labial involvement while in remission. A review of the literature is presented with special reference to the clinical characteristics of orofacial Crohn's disease and melkersson-rosenthal syndrome, which can mimic such presentation of Crohn's disease.
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keywords = labial
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8/9. Granulomatous cheilitis: a study of six cases.

    OBJECTIVE: Granulomatous cheilitis (GC) is a very rare disorder of unknown etiology. Clinically, GC is characterised by recurrent swelling of the labial tissues, which may be followed by a permanent enlargement. Histologically, the typical form of GC is characterised by the formation of scattered aggregates of non-caseating granulomas. GC is the most frequent sign of orofacial granulomatosis, a disorder under which also encompasses sarcoidosis, Crohn's disease, atypical tuberculosis, Anderson-fabry disease, possibly some allergic reactions, and melkersson-rosenthal syndrome (MRS). Some consider GC as an oligosymptomatic or monosymptomatic form of MRS. SUBJECTS AND methods: In this study we examined the clinical records of six patients presenting with GC which were examined and treated in the Department of oral medicine and pathology of the Dental School of Aristotle University of Thessaloniki (greece) during a 16-year period. In five of six patients a persistent swelling of the lower lip was recorded, one of whom also developed swelling in the upper lip. In one case the swelling was present in both lips and in another the GC was the only clinical finding, while in the other five cases it was accompanied by at least one other feature of MRS. In five cases, the histological picture revealed non-caseating granulomas. RESULTS: The treatment with the intralesional infusion of corticosteroids in three cases and the oral administration of corticosteroids in two cases was successful. One of the patients refused to be treated. This patient also presented later with permanent swelling of the upper lip.
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ranking = 1
keywords = labial
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9/9. melkersson-rosenthal syndrome: reduction cheiloplasty utilizing a transmodiolar labial suspension suture.

    melkersson-rosenthal syndrome is a rare condition which consists of progressive and recurrent orofacial edema, intermittent facial palsy, and lingua plicata. Labial swelling is the most common feature of this syndrome complex. The lips may enlarge up to three times their normal size, resulting in aesthetic deformity and functional disability. Traditional medical interventions are only marginally successful in treating this syndrome. We present a case of melkersson-rosenthal syndrome with massive lower lip enlargement, which was successfully treated by combining a fleur-de-lis resection with a transmodiolar labial suspension suture. Five-year follow-up photographs are presented.
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ranking = 5
keywords = labial
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