Cases reported "Mouth Diseases"

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1/33. Linear IgA disease histopathologically and clinically masquerading as lichen planus.

    In each of 2 cases reported, the patient presented with features of erosive lichen planus or lichenoid drug eruptions and an incisional biopsy taken from the patient was diagnosed histologically as lichen planus. Subsequent recurrences or exacerbations were associated with vesiculobullous lesions. Simultaneous or subsequent direct immunofluorescence studies--from the same tissue sample in one case and from a similar site in the other case--demonstrated classic features of linear IgA disease. Both patients were originally treated for lichen planus with systemic and/or topical corticosteroids with limited success. One patient was treated with sulfapyridine with minimal improvement. Both patients were subsequently treated with dapsone and demonstrated significant clinical improvement. We propose that linear IgA disease may be more common than reported in the oral cavity, inasmuch as many cases of recalcitrant lichen planus, erosive lichen planus, and lichenoid drug eruptions, especially those with a vesiculobullous component, may in reality represent linear IgA disease. We recommend that direct immunofluorescence be done in any case in which bullous lichen planus is suspected.
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2/33. oral manifestations of Schimmelpenning syndrome: case report and review of literature.

    Schimmelpenning syndrome (SS) is characterised by specific skin manifestations, skeletal defects, and central nervous system abnormalities. Here, the SS is briefly reviewed, and the oral and dental manifestations are described in a patient whose medical findings were previously published and included severe hypophosphatemic rickets. Significant oral and dental features included papillomatous lesions of the gingiva, hemihyperplasia (hemihypertrophy) of the tongue, bone cysts, aplasia of teeth, enlarged pulp chambers, hypoplastic or absent enamel, and an odontodysplasia-like permanent tooth.
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3/33. dyskeratosis congenita: report of a case.

    dyskeratosis congenita is a rare multisystem condition involving mainly the ectoderm. It is characterized by a triad of reticular skin pigmentation, nail dystrophy and leukoplakia of mucous membranes. Oral and dental abnormalities may also be present. Complications are a predisposition to malignancy and bone marrow involvement with pancytopenia. The case of a 14-year-old girl is described who presented with several of the characteristic systemic features of this condition, together with the following oral features: hypodontia, diminutive maxillary lateral incisors, delayed dental eruption, crowding in the maxillary premolar region, short roots, poor oral hygiene, gingival inflammation and bleeding, alveolar bone loss, caries and a smooth atrophic tongue with leukoplakia. Although this condition is rare, dental surgeons should be aware of the dental abnormalities that exist and the risk of malignant transformation within the areas of leukoplakia.
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4/33. Oral findings in Carpenter syndrome.

    Acrocephalopolysyndactyly Type II (Carpenter syndrome) is determined by autosomal recessive inheritance. Only some 40 cases have been described. Variable clinical signs have been described including prolonged retention of primary teeth and hypodontia. This paper describes the oral and dental findings in a family containing two affected brothers. The family pedigree is informative, as the mother has had children by three partners. The two affected individuals are full brothers. The first affected brother has delayed dental development, severe hypodontia and small tooth crown size. Mesio-distal and bucco-lingual dimensions were measured on the study models and compared with population data. The younger brother also has delayed dental development but only mild hypodontia. Their half sister has severe hypodontia but no signs of Carpenter syndrome. This family study demonstrates two affected individuals with typical clinical features and a pedigree compatible with autosomal recessive inheritance. Small tooth crown size has been shown by standardized measurement and evidence advanced that hypodontia is not part of the syndrome but a coincidental finding which segregates independently. We have also shown that the marked delay in emergence of teeth is associated more with problems of tooth eruption, possibly related to the bony abnormalities, than to a generalized delay in dental development.
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keywords = tooth, eruption
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5/33. Oral lichenoid reaction to imatinib (STI 571, Gleevec).

    Imatinib or STI 571 is a phenylaminopyrimidine derivative and a member of a new class of drugs known as signal transduction inhibitors. These compounds specifically inhibit the proliferation of v-abl- and bcr-abl-expressing cells and have recently been approved as treatment for chronic myeloid leukaemia (CML). Results have been promising, and imatinib may well be the best single agent for the treatment of CML in the near future. Here we report an erosive oral lichenoid eruption confined to the buccal mucosa and dorsum of the tongue which appeared 12 weeks after commencement of imatinib in a 72-year-old woman with CML. The histology was consistent with a lichenoid drug eruption. The lesions resolved upon withdrawal of the drug. To our knowledge, this is the first reported lichenoid reaction to imatinib, and in the setting of CML it must be differentiated from idiopathic lichen planus, paraneoplastic pemphigus and graft-versus-host disease.
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6/33. Oral-dental findings in dyskeratosis congenita.

    A 13-yr-old girl with dyskeratosis congenita is presented. Besides oral leukoplakia and nail dystrophies, there was evidence of pancytopenia, growth retardation, alopecia, mental retardation and microcephaly. The oral findings included caries, gingival recession, short-blunted roots, gingival bleeding, tooth mobility and severe alveolar bone loss resembling juvenile periodontitis.
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7/33. Cutaneous and oral eruption from oral exposure to nickel in dental braces.

    Oral eruptions due to nickel allergy are rare. A common presentation of intraoral contact dermatitis is the presence of lichenoid plaques on the buccal mucosa adjacent to the offending antigen. We report an unusual case of cutaneous and mucosal nickel allergy arising after placement of dental braces. An 11-year-old boy was referred by his orthodontist to the University of minnesota Occupational and Contact dermatitis Clinic to be evaluated for a possible metal allergy. The patient developed an itchy rash on his abdomen and under his wristwatch 1 week after dental braces were placed. He was diagnosed with allergic contact dermatitis from nickel. The patient avoided cutaneous nickel exposure and had a minimal resolution of his symptoms. One year later, the patient developed swelling and burning of the lips. Secondary to extreme discomfort, the braces, which contained nickel, titanium, and zinc, were removed. The patient underwent standard patch testing; the final reading at 96 hours showed a reaction to nickel, palladium, cobalt chloride, and neomycin. The patient experienced relief of his oral symptoms after removal of the braces. No current relevance to palladium, cobalt, or neomycin has been found.
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keywords = eruption
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8/33. Oral lichenoid eruption secondary to imatinib (Glivec).

    BACKGROUND: Oral lichenoid drug eruption (OLE) is a self-restricted inflammatory condition usually secondary to a variety of drugs and dental materials (mercury, gold), and rarely to immunomodulators and new antineoplastic agents such as imatinib mesylate (Glivec). OLE clinical features are reminiscent of lichen planus and include symmetric and asymptomatic, erythematous, reticulated and painful ulcerated plaques. Histological examination reveals perivascular lymphocytic infiltrate and the presence of eosinophils that obscures the dermal-epidermal junction, with acanthosis and mild hyperkeratosis. methods: A 62-year-old man was treated with Glivec for metastatic gastrointestinal stromal tumour (GIST). RESULTS: The patient presented with grey-violaceous plaques on both cheek mucosal surfaces and the lateral sides of the tongue and labial mucosa, with a reticular pattern resembling oral lichen planus. These lesions appeared approximately 1 year after therapy and partially subsided in about 10 weeks, to clear after topical corticosteroids and oral omeprazole. The diagnosis of OLE induced by imatinib was confirmed by histological biopsy evaluation; immunohistochemical analysis revealed intense immunoreactivity with only cytokeratins 5/6 and 14 in the epithelium of affected mucosa. All laboratory investigations proved asiderotic anaemia but excluded infectious causes. CONCLUSION: It is believed that this patient developed imatinib-induced OLE; this relationship with therapy rather than the underlying disease rules out a paraneoplastic reaction, and negative immunofluorescence excluded autoimmune dermatosis. In addition, we propose that these lesions are correlated with abnormal expression of some cytokeratins directly caused by this drug.
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keywords = eruption
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9/33. A case of symptomatic primary HIV infection.

    A 30-year-old homosexual Japanese man had fourteen days of fever, malaise, appetite loss, sore throat, and four days of diarrhea and slightly congested eyes before he developed a skin eruption. He presented with measles-like exanthems on his face, trunk, and extremities. Deep red enanthems were seen on his left buccal mucosa opposite the premolar teeth, and whitish enanthems were seen on the buccal and gingival mucosa. HIV rna was detected at the high concentration of 5.8 x 10(6) copies /ml in his serum. cerebrospinal fluid examination revealed aseptic meningitis with 5,488 copies /ml of HIV rna. Anti-HIV 1 antibodies against Gp160 and p24 tested by Western blot assay showed seroconversion on day 5 of his admission, seven days after he developed the skin eruptions. The fever lasted for three weeks from the initial onset, and the skin eruptions lasted for twelve days. Histopathologically, a mononuclear cell infiltration was seen mainly in the upper dermis surrounding small vessels and sweat ducts, with CD8 cytotoxic T lymphocytes predominant. Additionally, CD1a putative interdigitating dendritic cells had also infiltrated perivascularly, and were surrounded by CD8 and CD4 T cells. in situ hybridization study failed to detect HIV products in skin biopsy specimens. Our findings suggested that CD8 T cells and their interaction with CD1a dendritic cells in the skin may be important in inducing skin manifestations in acute hiv infections.
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ranking = 1.5
keywords = eruption
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10/33. Modification of the maxillary Le Fort I osteotomy in cleft-orthognathic surgery: the unilateral cleft lip and palate deformity.

    Modifications of the Le Fort I osteotomy are described that allow for the simultaneous routine and safe management of maxillary hypoplasia, residual oronasal fistula, bony defects, soft-tissue scarring, and cleft-dental gap in adolescents with unilateral cleft lip and palate (UCLP). The results of this operation with 40 consecutive patients are presented, together with follow-up findings ranging from 15 months to 4 years 5 months. Parameters reviewed include cleft-dental gap closure, maintenance of attached gingiva at the cleft site, maintenance of a positive overjet and overbite, closure of residual oronasal fistula, the need for prosthetics to complete dental rehabilitation, and surgical morbidity. Thirteen of the patients also underwent simultaneous sagittal split osteotomies of the mandible, and 29 had a genioplasty performed. In 32 of the patients surgical cleft-dental gap closure was planned, and was successfully executed in all but one. Thirty-seven patients underwent successful simultaneous oronasal fistula closure, but in three cases, small residual fistulas remained. In all cases, attached gingiva was maintained in the region of the cleft site and along the tooth-bearing surfaces. Almost all of the patients maintained a positive overjet (39 of 40) and 85% maintained a positive (34 of 40) or at least neutral (4 of 10) overbite. Complications were few and generally not serious.
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keywords = tooth
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