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PubMed2005

Juvenile hyaline fibromatosis: a case report.

Juvenile hyaline fibromatosis ( JHF ) is a rare autosomal recessive disease characterized by papulonodular skin lesions, gingival hyperplasia, joint contractures, and bone lesions. The skin lesions may consist of multiple large tumors, commonly on the scalp and around the neck, and small pearly, pink papules and plaques on the trunk, chin, ears, and around the nostrils. Here, we report a 2-year-old boy with characteristic stiffness of the knees and elbows and pink confluent papules on the paranasal folds, and periauricular and perianal regions. He also had hard nodules all over the scalp and around the mouth, and severe gingival hyperplasia. The lesions were totally excised and clinicopathological diagnosis was JHF.

Child, Preschool↗
PubMed1997

Severe bleeding from a pregnancy tumor. A case report.

BACKGROUND: Hyperplastic gingivitis and gingival hyperplasia accompanying gestation have been termed "pregnancy gingivitis" and "pregnancy tumor." The condition is benign but, rarely, is complicated by severe bleeding that is difficult to manage. A single case of a pregnancy tumor is reported. CASE: A 28-year-old woman in the third trimester was evaluated for treatment of a pregnancy granuloma with recurrent episodes of severe bleeding for two weeks. Conservative management by firm pressure on gauze packs was applied to control the bleeding, but in vain. Induction of labor was conducted at 37 weeks partially because of term pregnancy and uncontrollable bleeding from the gingiva. An emergency cesarean section was done because of acute fetal distress during induction of labor. A healthy infant was delivered. The gingival bleeding stopped spontaneously five days afterwards. The patient was given thorough dental prophylaxis and oral hygiene instructions. The buccal granulomatous tumor was decreased in size four weeks postpartum. CONCLUSION: Careful oral dental hygiene, removal of dental plaque and debris, and use of soft toothbrushes are important during pregnancy to avoid occurrence of a pregnancy tumor. If uncontrolled bleeding occurs, management should be based on the individual condition and should range from supportive therapy--such as desiccation of bleeders; local, firm compression and oral hygiene to blood transfusion--as well as medication to accelerate fetal lung maturity or even termination of pregnancy to save the patient's life, as with treatment of uncontrollable eclampsia.

Adult↗
PubMed1977

[Hormonal contraceptives and periodontium].

The clinical changes of the gingiva reported in the literature (gingival hyperplasia and new growths like the pregnancy epulis) in connection with oral hormonal contraception as well as the causal connection between sexual hormone activity and the changes of gingiva and oral mucosa are discussed. Our own experiments show that the increase in the sulcus fluid flow rate under administration of combination preparations is largely independent of an increase in the degree of gingival inflammation.

Adult↗
PubMed1989

Focal dermal hypoplasia. Current concepts and differential diagnosis.

Focal dermal hypoplasia syndrome (FDH) is a congenital disorder of mesodermal derivation with some ectodermal participation. The disorder, first described in 1962, is characterized by tenacious defects consisting of thinning of the skin; herniations of adipose tissue; abnormal skin pigmentation and other skeletal, dental, ocular, hair, and nail abnormalities. A rare instance of focal dermal hypoplasia syndrome is reported as characterized by papillary gingival hyperplasia. Differential diagnostic guidelines that separate focal dermal hypoplasia from a host of other skin and mucous membrane disorders are discussed along with therapeutic modalities and the possible relationship of human papillomavirus to focal dermal hypoplasia syndrome.

Adult↗
PubMed1992

[Drug-induced changes in the teeth and mouth. II].

As in the previous paper the unwanted effects of drugs or chemicals in the orofacial region are described. The authors take into consideration alterations such as gingival hyperplasia and hypertrophy, discoloration of the oral mucosa and teeth, oral ulceration and stomatitis, cervical lymphadenopathy, drug induced blood dyscrasias, bleeding caused by aspirin and other drugs, and cleft lip and cleft palate.

Cleft Lip↗
PubMed1979

[Hydantoin hyperplasia--the interesting case].

The author describes a case of excessive gingival hyperplasia in a 9-year-old girl under hydantoin medication. The hyperplasia of the gingivae inhibited the regular uptake of food and the eruption of the permanent teeth.

Child↗
PubMed2005

A solitary calvarial lytic lesion with typical histopathological findings of juvenile hyaline fibromatosis.

Juvenile hyaline fibromatosis (JHF) is a rare systemic disease characterized by papulonodular skin lesions, gingival hyperplasia, joint contractures, and osteolytic lesions on long bones and the skull. It has recently been reported that the disease is caused by mutations in the gene encoding capillary morphogenesis protein-2 (CMG-2). To date, fewer than 60 cases have been published in the literature. Partial disease expression is common, but no cases featuring a solitary calvarial lesion have been reported. The authors discuss this 4-year-old boy with a solitary calvarial osteolytic lesion whose histopathological examination exhibited findings characteristic of JHF. Mutational analysis, however, revealed that there were no mutations in the CMG-2 gene. Two years after surgery, he was free of any complaints as well as gingival hyperplasia, joint contractures, and new skull or skin lesions. This patient's condition may represent clinical or genetic heterogeneity associated with JHF. Whether solitary lesions mimicking JHF can arise from somatic mutation of the CMG-2 gene remains to be proven.

Child, Preschool↗
PubMed1993

Immunohistochemical distribution of extracellular matrix proteins as a diagnostic parameter in healthy and diseased gingiva.

This study showed histopathological findings of the extracellular matrix in healthy, inflamed, and hyperplastic human gingiva with immunohistochemical techniques. The distribution of collagen types V and VI, as well as of glycoprotein fibronectin, shows that they are extracellular matrix structural components which differentiate the tissue pathology. The orientation of the collagen fibers, the intensity of the fluorescent staining, the thickness of the fibrillar component, and the topographical localization of the connective tissue proteins are important parameters for tissue morphology. Therefore, bacterial deposits and the pharmacodynamic properties of drugs associated with gingival hyperplasia lead to an alteration of the matrix compared to the healthy tissues. This may be important in the tissue pathology in cases when the medical history of the patient is not known, as well as in forensic medicine.

Adult↗
PubMed1988

[Maxillofacial manifestations of Cowden's disease. Apropos of 2 cases].

Two new cases of Cowden's disease observed in two young sisters are reported. The diagnosis was based on the clinical observation of oral mucosa lesions (gingival hyperplasia, papillomatosis, scrotal tongue). In one case, visceral manifestations in childhood were observed. Cowden's disease is a familial affection (autosomal dominant trait) characterized by association of oral mucosa and skin lesions and of multiple hamartomas involving glandular tissues. Clinical and familial history investigations are justified by the high risk of malignant tumors (breast carcinoma).

Adolescent↗
PubMed1988

Drugs and the periodontium.

The periodontium and periodontal disease activity can be affected by systemic drug therapy. Many drugs can have an adverse effect on the periodontium, i.e., gingival hyperplasia. Alternatively, some drugs can modify the inflammatory and immunological responses of the periodontal tissues to bacterial plaque. The aim of this review is to evaluate the effects of drug therapy on the periodontium and periodontal disease activity, and where possible, to relate such changes to the pharmacodynamics of the drugs considered. Drugs which have been reported to affect the periodontium can be categorised as follows: anti-epileptics, immunosuppressants, corticosteroids, non-steroidal anti-inflammatory drugs and hormones. Those drugs whose pharmacodynamics are clearly established and which affect the rate of periodontal disease activity, may provide information on the mechanisms of periodontal destruction. Finally, the mechanisms of drug-induced gingival hyperplasia (overgrowth) are discussed in relationship to the drugs' pharmacodynamics and pharmacokinetics.

Animals↗
PubMed2002

Oral myofibromatosis: an unusual cause of gingival overgrowth.

BACKGROUND: This case report describes a rare benign tumour, which presented as discrete areas of gingival hyperplasia affecting both the mandible and the maxilla. METHOD: Surgical excision of the lesions was carried out under local anaesthetic. Histopathological examination confirmed the diagnosis of oral myofibromatosis. RESULTS: The condition responded to surgical excision and appears to have limited growth potential. It affects a wide spectrum of ages and can be alarming due to rapid enlargement and ulceration, so careful diagnosis is important to avoid unnecessary aggressive treatment.

Actins↗
PubMed2001

Long-term treatment of focal segmental glomerulosclerosis in children with cyclosporine given as a single daily dose.

Cyclosporine (CsA) has been successfully used for treatment of children with focal segmental glomerulosclerosis (FSGS) and nephrotic syndrome (NS) for the last decade. Response rates of 50% to 100% have been reported using twice-daily dosing of 5 to 32 mg/kg/d, achieving trough blood levels of 70 to 500 ng/mL. Treatment has been associated with a high incidence of side effects, including nephrotoxicity, hypertension, gingival hyperplasia, and hirsutism. To determine whether once-daily low-dose CsA could minimize side effects and still induce remission, 21 children with biopsy-proven FSGS and NS, each treated with CsA, 4.6 +/- 0.8 mg/kg/d, with no predetermined target trough blood levels, were studied. Eleven of 21 children (52%) attained complete remission and 5 of 21 children (24%) attained partial remission, for a total response rate of 76%. Mean time to response was 2.8 +/- 0.8 months, and mean duration of therapy was 20.6 +/- 13.7 months. CsA dosage was tapered or stopped in 9 responders; 3 of these patients maintained remission at last follow-up 6 to 13 months later, and 6 patients relapsed at 1.5 to 18.7 months (mean, 8.7 months). Five of these 6 patients responded again when CsA therapy was restarted or the dosage was increased. Twelve of 16 responders were still being administered CsA at last follow-up 11 to 60 months (mean, 24.6 months) later. Five of 21 patients (24%) had no response to CsA during 2 to 27 months of therapy; 4 of these 5 patients developed end-stage renal disease after CsA therapy was stopped. Side effects of CsA therapy were minimal: 1 patient each developed new-onset hypertension or gingival hyperplasia, and no patient had hirsutism or nephrotoxicity. Single daily low-dose CsA appears to be effective for long-term treatment of children with FSGS and NS, with fewer side effects than twice-daily dosing.

Adolescent↗
PubMed1987

[Periodontal diseases in adults. Immunohistochemical study of inflammatory cells and of collagen].

An immunohistochemical study was performed on 9 gingival biopsy specimens from patients suffering of a periodontal disease comparatively to 7 control specimens from patients with inflammatory gingival hyperplasia of various causes and from healthy gums. We used mono or polyclonal antibodies as markers of the different types of inflammatory cells (T-1 lymphocytes and their subsets, B-lymphocytes, macrophages, plasma cells). In patients with periodontal disease, the high percentage of transformed T-lymphocytes with a majority of helpers was obvious. In controls, the inflammatory cells were principally B-lymphocytes, macrophages and plasma cells. The characterization of different types of collagen produced by periodontal fibroblasts was performed by specific antibodies (anti I, III, V collagen sera). The respective ratio and the topographic distribution of those collagen substances were different in patients with periodontal disease and controls. In the first eventuality, one could find quite exclusively type I collagen. In the other group, type I collagen was associated with type III sub epithelial and type IV perivasor ar collagen. By means of these findings, the pathogenesis of the periodontal disease was discussed and its immune mechanism was suggested.

Biopsy↗
PubMedDate not supplied

Oral manifestations of drug therapy.

Increasingly, patients with complicated medical problems are coming to dental offices for treatment. These patients report using a variety of medications to control one or more chronic conditions. Many of these medications produce changes in the mouth because of toxic overdoses, side effects, allergic reactions, or as a consequence of the primary action of the drug. These effects include: xerostomia, gingival hyperplasia, candidiasis, mucositis, stomatopyrosis, gingival bleeding, petechia, salivary gland problems, intrinsic stain, cheilitis, erythema multiforme, lichen planus, ulcerations, taste changes, tardive dyskinesias, and soft tissue pigmentation. This article discusses presentations and causes of oral changes secondary to systemic drug use.

Drug-Related Side Effects and Adverse Reactions↗
PubMed1997

Collagen synthesis in idiopathic and dilantin-induced gingival fibromatosis.

Gingival fibromatosis is an uncommon but benign oral disease which causes progressive and non-hemorrhagic enlargement of the gingiva. There are two types of gingival fibromatosis: idiopathic gingival fibromatosis (GF), which is of unknown cause and may be associated with hereditary factors, and drug-induced gingival fibromatosis which is caused primarily by phenytoin intake. In cases of gingival fibromatosis, either the teeth are delayed in emergence or most of the crowns are embedded in the soft tissue even after full eruption. The objective of this study was to examine the basis of excess collagen formation in the two types of gingival fibromatosis in four patients admitted to the dental clinic at Kaohsiung Medical College Hospital, Kaohsiung, Taiwan. There were two male patients, aged ten (Case 1) and sixteen (Case 2), with idiopathic gingival fibromatosis and hypertrichosis, and two female patients, aged sixteen (Case 3) and eleven (Case 4), with Dilantin-induced gingival hyperplasia (DGH). Cultures of gingival fibroblast were established either from clinically excised hyperplastic tissues or from pre-orthodontic surgical normal gingiva. The synthesis of collagen and levels of prolyl hydroxylase, a key enzyme in collagen synthesis, were examined in the healthy and affected fibroblasts. Our studies showed that after two days in culture, fibroblasts from all four patients multiplied faster than healthy gingival fibroblasts, though the amount of DNA and protein per cell remained unchanged. In addition, all cultures (except Case 1) had a 2- to 3- fold increase of prolyl hydroxylase activity over that of the controls. As in the cases of prolyl hydroxylase activity, Case 1 did not show any change in collagen synthesis when compared to the control. However, Cases 2, 3, and 4 showed appreciable collagen increases in the cell and medium: 61% and 60% for Case 2; 16% and 36% for Case 3; and 21% and 80.7% for Case 4 respectively.

Adolescent↗