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1/29. Histopathology and electron and immunofluorescence microscopy of gingivitis granulomatosa associated with glossitis and cheilitis in a case of Anderson-fabry disease.

    A 17-year-old white boy with signs, symptoms, and family history of angiokeratoma corporis diffusum universale, Anderson-fabry disease (AFD), developed recurrent and then persistent swelling of both lips, erythematous hyperplastic gingivae, and a pebbled tongue. Positive blood findings were raised serum IgE, decreased T-cell level, and increased B-cell level. Histopathology of the gingiva showed noncaseating granulomas with multinucleate giant cells containing Schaumann bodies and large plasma-cell infiltrates in which immunofluorescence demonstrated immune globulins of several classes. Electron microscopy and histochemistry demonstrated ceramide in the vasculature. No glycolipid was found in the macrophages or giant cells of the granulomas which, in contrast, resembled sarcoid reactions. plasma cells with Russell bodies and immune reaction-induced degranulation of mast cells were also identified. The pathogenesis of the oral findings possibly relates to altered immune reactivity associated with damage to the microvasculature analogous to that in melkersson-rosenthal syndrome.
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2/29. melkersson-rosenthal syndrome: review of the literature and case report of a 10-year misdiagnosis.

    melkersson-rosenthal syndrome is classically described as a triad of orofacial swelling, facial palsy, and fissured tongue. More often this syndrome presents in its oligosymptomatic forms. melkersson-rosenthal syndrome may not be as rare as suspected but rather a syndrome that often goes undiagnosed. Presented is the case of a mentally challenged man who was eventually diagnosed with melkersson-rosenthal syndrome only after being misdiagnosed and incorrectly treated for an odontogenic infection for more than a decade.
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3/29. melkersson-rosenthal syndrome in childhood: successful management with combination steroid and minocycline therapy.

    The melkersson-rosenthal syndrome consists of a triad of recurrent lip and/or face swelling, fissured tongue, and intermittent facial palsy. Onset of the symptoms may occur during childhood, and treatment of the condition is difficult. We describe two children with melkersson-rosenthal syndrome in whom combination treatment with prednisone and minocycline proved effective and well tolerated.
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4/29. melkersson-rosenthal syndrome: report of three cases.

    melkersson-rosenthal syndrome is a rare disorder consisting of the triad of persistent or recurrent orofacial edema, relapsing facial paralysis and fissured tongue. It is far more uncommon to find the complete triad since it generally presents in oligosymptomatic forms. We present three cases of the melkersson-rosenthal syndrome with the classic triad of symptoms and discuss the etiology and the clinical and electromyography findings of this syndrome.
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5/29. Therapeutic response to thalidomide in melkersson-rosenthal syndrome: a case report.

    BACKGROUND: melkersson-rosenthal syndrome (MRS) is a rare disorder of unknown etiology characterized by a triad of symptoms: recurrent orofacial swelling, relapsing facial palsy. and a fissured tongue. A differential diagnosis must be made with other granulomatous diseases, such as sarcoidosis and oral crohn disease; however, the histologic findings of noncaseating, sarcoidal granulomas support the diagnosis of MRS. RESULTS: Many therapeutic modalities have been described for this disease. In this case report, we present a patient with MRS that was treated with thalidomide because of the identification of tumor necrosis factor a in the lesion by immunohistochemical analysis. This is the first reported detection of tumor necrosis factor a in an MRS lesion, as well the first reported use of thalidomide to treat this clinical condition.
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6/29. melkersson-rosenthal syndrome in the periocular area: a review of the literature and case report.

    A triad of facial palsy, facial edema, and furrowed tongue characterizes melkersson-rosenthal syndrome, a rare, noncaseating granulomatous disease of unknown cause. Although most reported cases of melkersson-rosenthal syndrome involve swelling of the perioral area, the authors present a case of melkersson-rosenthal syndrome involving the periocular area. Because of its rarity, the syndrome is usually ignored and misdiagnosed; however, the syndrome should not only be considered in the classic perioral presentation but also in the rare periocular form, which may be confused with orbital tumors and orbital pseudotumors. Biopsies should be performed routinely in all patients who present with eyelid edema of unknown etiology. The physician and surgeon who see patients with head and neck pathology should be familiar with melkersson-rosenthal syndrome, and with the possibility of its presentation in the orbit and periocular region.
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7/29. Treatment and follow-up of persistent granulomatous cheilitis with intralesional steroid and metronidazole.

    Granulomatous cheilitis (GC) is a chronic edema which frequently affects the upper lip due to granulomatous inflammation. Its etiology is currently unknown. This rare disease is generally accompanied by melkersson-rosenthal syndrome (MRS), characterized by scrotal tongue, orofacial edema and facial paralysis. However, it is also known to develop only with orofacial edema. Granulomatous cheilitis is a difficult disease to treat because of recurrences. There are contradictory reports about the results of treatment without surgical intervention and the rates of recurrence. Our case was a 57-year-old female patient who was characterized by orofacial edema only. The edema and erythema had persisted for 1 year before admission. In the present case, application of intralesional corticosteroid treatment as a total of three injections over 3 consecutive months (one injection per month) and the accompanying metronidazole treatment brought about successful results. No recurrence was observed in the follow-up.
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8/29. The surgical management of melkersson-rosenthal syndrome.

    A triad of facial palsy, orofacial edema, and furrowed tongue constitutes an uncommon condition known as melkersson-rosenthal syndrome (MRS). We report on 14 patients with melkersson-rosenthal syndrome. Two patients had facial palsy, 12 had orofacial edema, and 1 patient had a furrowed tongue. Nine patients were treated medically. Intralesional steroid therapy had a 75 percent recurrence rate. Systemic steroid therapy resulted in remission in two of three patients. Surgical excision and reconstruction were carried out in five patients with chronic lip or eyelid edema. This provided relief in all cases. The etiology, clinical presentation, and histologic features are discussed. Three illustrative cases are presented. An algorithm is provided that guides the surgeon with regard to both the medical and surgical treatment of the patient with melkersson-rosenthal syndrome.
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9/29. Persistent unilateral orbital and eyelid oedema as a manifestation of melkersson-rosenthal syndrome.

    melkersson-rosenthal syndrome (MRS) is a complex neuromucocutaneous disorder characterized by localized orofacial oedema and cranial nerve dysfunction, frequently associated with minor signs, including furrowed tongue. Complete forms are rare whereas mono- and oligosymptomatic variants are more common. A 71-year-old man presented with a 2-year history of relapsing and progressively persistent oedema of the right eyelids and periorbital region. A fissured tongue and telangiectatic rosacea had been present since the age of 50 and 60 years, respectively. The patient was also affected by essential hypertension and diabetes mellitus. A skin biopsy showed a marked upper dermal oedema, and small epithelioid cell granulomas arranged in perivascular and perilymphatic location. collections of small epithelioid cells were occasionally observed within lymphatic spaces. No acid-fast bacteria, fungi or foreign bodies were detected. Intralesional corticosteroids induced transient improvement, whereas minocycline, clofazimine and dapsone have been ineffective. MRS may present with unilateral eyelid and periorbital swelling. Differential diagnoses of such cases may include a variety of cutaneous, ophthalmic and systemic diseases.
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10/29. clofazimine as elective treatment for granulomatous cheilitis.

    cheilitis granulomatosa (CG) is a rare, idiopathic inflammatory disorder that usually affects young adults and clinically is characterized by diffuse, non-tender, soft to firm swelling of one or both lips. A variant of granulomatous cheilitis is melkersson-rosenthal syndrome when associated with facial paralysis and furrowed tongue. Several treatments have been used with variable success. We report 3 cases of GC treated with oral clofazimine 100 to 200 mg daily for 3 to 6 months obtaining regression of lesions in all treated cases. hyperpigmentation and elevation of liver enzymes were observed as side effects.
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