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PubMed2003

Calcospherules associated with juvenile hyaline fibromatosis.

Juvenile hyaline fibromatosis is a rare autosomal recessive genodermatosis that manifests with 1 to 5 cm skin tumors, gingival hyperplasia, osteolytic bone lesions, and joint contractures. The skin tumors are most often located on the head and periarticular extremities. A case of juvenile hyaline fibromatosis with typical skin tumors that on biopsy exhibited basophilic calcospherules is presented. Although noted in a single case, these structures may be a novel, yet helpful, diagnostic marker.

Adult↗

PubMed2005

Unusual oral manifestations and evolution in glycogen storage disease type Ib.

Glycogen storage disease type Ib is a rare inherited metabolic disorder that is caused by a deficiency of glucose-6-phosphate translocase with consequent accumulation of glycogen. The purpose of this study is to report a case affected by glycogen storage disease type Ib in which unusual oral findings were evident and to review the pertinent literature. The disease presents with failure to thrive, hepatomegaly, hypoglycemia, hyperlacticacidemia, neutropenia, and neutrophilic dysfunction causing increased susceptibility to recurrent infections. Common intraoral manifestations are dental caries, gingivitis, periodontal disease, delayed dental maturation and eruption, oral bleeding diathesis, and oral ulcers. Conversely, unusual oral lesions were observed in this case as hyperplastic-hypertrophic gingiva and giant cell granulomatous epulis. The treatment with granulocyte colony-stimulating factor markedly increased the neutrophil counts and reduced the frequency of infections and inflammations. Proper evaluation of the patient's oral condition, a program of preventive measures, and suitable medical consultation are important to minimize and avoid long-term complications.

Adolescent↗

PubMed1985

Crohn's disease limited to the mouth and anus.

A young girl developed painful swelling and ulceration of her lips and mouth. She had gingival hyperplasia, an anal fissure, and anal skin tags without evidence of intervening intestinal disease. A diagnosis of Crohn's disease was made. All lesions regressed with appropriate therapy. She remains asymptomatic 14 months after presentation.

Adolescent↗

PubMed1997

Multicenter randomized trial comparing tacrolimus (FK506) and cyclosporine in the prevention of renal allograft rejection: a report of the European Tacrolimus Multicenter Renal Study Group.

BACKGROUND: To confirm the results of a number of studies conducted in Europe, the United States, and Japan, this multicenter, randomized trial compared the 12-month efficacy and safety of tacrolimus- and cyclosporine-based immunosuppressive regimens in the prevention of renal allograft rejection. METHODS: A total of 448 renal transplant recipients were recruited from 15 centers and assigned to receive triple-drug therapy consisting of tacrolimus (n=303) or cyclosporine (n=145) in conjunction with azathioprine and low-dose corticosteroids. RESULTS: At 12 months after transplantation, tacrolimus therapy was associated with a significant reduction in the frequency of both acute (tacrolimus 25.9% vs. cyclosporine 45.7%; P<0.001 [absolute difference: 19.8%, 95% confidence interval: 10.0-29.6%]) and corticosteroid-resistant rejection (11.3% vs. 21.6%; P=0.001 [absolute difference: 10.3%, 95% confidence interval: 2.5-18.2%]). Actuarial 1-year patient (tacrolimus 93.0% vs. cyclosporine 96.5%; P=0.140) and graft survival rates (82.5% vs. 86.2%; P=0.380) did not differ significantly between the two treatment groups. Overall, the safety profiles of the tacrolimus- and cyclosporine-based regimens were quite comparable. Infections, renal impairment, neurological complications, and gastrointestinal complaints were frequently reported but were mostly reversible in both groups. Higher incidences of elevated serum creatinine, tremor, diarrhea, hyperglycemia, diabetes mellitus, and angina pectoris were reported in the tacrolimus treatment group, whereas acne, arrhythmia, gingival hyperplasia, and hirsutism were more frequent with cyclosporine treatment. CONCLUSIONS: The significant reduction in the incidence of episodes of allograft rejection observed with tacrolimus therapy may have important long-term implications given the prognostic influence of rejection on graft survival.

Adolescent↗

PubMed1997

Assessment of health-related quality of life in kidney transplant patients receiving tacrolimus (FK506)-based versus cyclosporine-based immunosuppression. FK506 Kidney Transplant Study Group.

BACKGROUND: We evaluated health-related quality of life (HQL) in kidney transplant patients participating in a multicenter, prospective, randomized, phase III trial comparing tacrolimus to cyclosporine. HQL data were available for 303 of 412 patients and assessed with the SF-36 Health Survey and six multi-item scales: Current Health, Health Outlook, Health Distress, Fleming Self-Esteem, Bergner Physical Appearance, and Sexual Functioning. METHODS: Patients completed surveys at baseline, week 6, and months 3, 6, and 12. The mean change in HQL was evaluated by rejection occurrence and number of hospitalizations. Analysis of covariance was used to model endpoint HQL scores as a function of treatment group and baseline HQL. RESULTS: All scales but two met psychometric standards for group-level comparisons. Baseline demographics and HQL scores were not different by treatment. The mean HQL change was lower for patients with rejection compared with no rejection in seven of eight SF-36 scales and three of four remaining supplemental scales. One year after transplantation, study patients were functioning at least as well as half of the general population in Vitality and Role-Emotional Functioning, moving from the 18th percentile of the U.S. population scores to the 50th percentile for Vitality and 54th percentile for Role-Emotional Functioning. Patients improved their percentile ranking by at least 20 points in five of eight SF-36 scales. CONCLUSIONS: Patients with kidney disease demonstrate substantial HQL burden before transplantation, and transplantation is associated with substantial HQL improvements. Rejection is associated with less HQL improvement. Endpoint HQL values were significantly different (P<0.05) by treatment, favoring tacrolimus, in the Bergner Physical Appearance scale, which was designed to measure the HQL impact of side effects such as gingival hyperplasia and facial hirsutism on physical appearance.

Adolescent↗

PubMed1999

Sirolimus (rapamycin)-based therapy in human renal transplantation: similar efficacy and different toxicity compared with cyclosporine. Sirolimus European Renal Transplant Study Group.

BACKGROUND: Sirolimus (rapamycin) is a potent immunosuppressant with a mechanism of action different from cyclosporine (CsA) or tacrolimus. METHODS: In 11 European centers, first cadaveric renal allograft recipients were randomized to CsA (n=42) or sirolimus (n=41). Dosing of these agents was concentration-controlled and open-labeled. All patients received corticosteroids and azathioprine. RESULTS: At 12 months, graft survival (98% sirolimus vs. 90% CsA), patient survival (100% vs. 98%), and incidence of biopsy-confirmed acute rejection (41% vs. 38%) were similar. Serum creatinine was lower with sirolimus, significantly (P< or =0.05) so at 3 and 4 months, and serum uric acid and magnesium were normal. Laboratory abnormalities reported significantly more often with sirolimus included hypertriglyceridemia (51% vs. 12%), hypercholesterolemia (44% vs. 14%), thrombocytopenia (37% vs. 0%), leukopenia (39% vs. 14%), and, of lesser importance, increased liver enzymes and hypokalemia. These abnormalities improved 2 months after transplantation when the sirolimus target trough level was lowered from 30 to 15 ng/ml. Occurrence of cytomegalovirus was comparable (14% vs. 12%); incidences of herpes simplex (24% vs. 10%, P=0.08) and pneumonia (17% vs. 2%, P=0.03) were higher with sirolimus. No gingival hyperplasia was seen with sirolimus, tremor was rare, and hypertension was less frequent (17% vs. 33%). Two malignancies were observed with CsA and none with sirolimus. CONCLUSIONS: Results at 12 months suggest that sirolimus can be used as base therapy in the prophylaxis of acute renal transplant rejection, and has a safety profile that differs from CsA.

Adult↗

PubMed2004

Long-term benefits with sirolimus-based therapy after early cyclosporine withdrawal.

Graft function at 6 or 12 mo is positively correlated with renal transplant survival. The 36-mo results of a study that tested whether withdrawing cyclosporine (CsA) from a sirolimus (SRL)-CsA-steroid (ST) regimen would affect renal graft survival are reported. Eligible patients (n = 430) who were receiving SRL-CsA-ST were randomly assigned at 3 mo to remain on SRL-CsA-ST or to have CsA withdrawn (SRL-ST group). At 36 mo, the calculated GFR was significantly better with SRL-ST (47.3 versus 59.4 ml/min; P < 0.001) as was the slope of the GFR (-3.6 versus 0.8 ml/min; P < 0.001). This was accompanied by growing trend for improved graft survival in the SRL-ST group (85.1% versus 91.2%, P = 0.052 at 36 mo; 81.4% versus 91.2%, P = 0.015 in a cumulative data analysis up to 54 mo), despite numerically more biopsy-proven acute rejections after randomization (5.6% versus 10.2%; P = 0.107). Lipid parameters were similar between groups, whereas both systolic and diastolic BP were significantly lower in the SRL-ST group. Investigator-reported hypertension, abnormal kidney function, edema, hyperuricemia, hyperkalemia, gingival hyperplasia, and Herpes zoster occurred significantly more often in SRL-CsA-ST patients. Abnormal liver function test results, hypokalemia, thrombocytopenia, and abnormal healing were reported significantly more often with SRL-ST. The discontinuation rate was significantly higher for SRL-CsA-ST (48% versus 38%; P = 0.041). In conclusion, withdrawing CsA from a SRL-CsA-ST regimen at 3 mo after transplantation resulted in long-term benefits for renal transplant recipients.

Acute Disease↗

PubMed2003

Common oral manifestations during pregnancy: a review.

UNLABELLED: Pregnancy has far-reaching systemic effects extending beyond the reproductive system. Oral and masticatory changes during pregnancy have been documented for many years; however, their magnitude and frequency have not been stressed. This review highlights the major oral complications during pregnancy. Pregnancy gingivitis and pregnancy tumor are described and presented, and possible preventive strategies are suggested. TARGET AUDIENCE: Obstetricians & Gynecologists, Family Physicians. LEARNING OBJECTIVES: After completion of this article, the reader will be able to summarize the major oral complications during pregnancy, to outline the etiologic factors associated with each of these conditions, and to explain the role of dental treatment during pregnancy.

Dental Care↗

PubMedDate not supplied

Infantile systemic hyalinosis or juvenile hyaline fibromatosis?

Infantile systemic hyalinosis and juvenile hyaline fibromatosis are presumably autosomal recessive inherited diseases of unknown origin in which accumulation of an amorphous, hyaline material occurs in the skin and other organs. Both disorders may show clinical overlapping, suggesting that they might represent different variants of the same disease spectrum. We describe a 6-year-old boy with such overlap. Salient features included papular skin lesions on his face and neck, gingival hyperplasia, perianal nodules, large subcutaneous tumors on the scalp, hyperpigmented plaques over the metacarpophalangeal joints and malleoli, limited joint movement, diffuse osteopenia, short stature, and persistent diarrhea. Histopathologic and ultrastructural studies confirmed the presence of hyalin material in the dermis. The term systemic hyalinosis involves both conditions and should be preferred until a clear distinction can be made between them.

Child, Preschool↗

PubMed1990

Side-effect profile of cyclosporin A in patients treated for psoriasis.

This is a review of the side-effects of cyclosporin A (CyA) in patients with severe psoriasis; renal dysfunction and hypertension are discussed elsewhere. In particular, paraesthesia, hypertrichosis, gingival hyperplasia and gastrointestinal disorders may occur, but are generally transient, mild-to-moderate in severity and only rarely require discontinuation of CyA. Infections are not a problem. As expected with an immunosuppressive drug, there is the possible risk of tumour development, particularly squamous cell carcinomas. However, these skin malignancies developed almost exclusively in patients previously treated with PUVA and/or methotrexate. The few lymphoproliferative disorders regressed spontaneously on discontinuation of the drug. Whether the isolated cases of solid tumours were CyA-related is not known. Apart from a raised serum creatinine, an important indicator of renal dysfunction, the laboratory abnormalities included hypomagnesaemia, hyperkalaemia, increased uric acid, changes in liver function tests, and fluctuations in the serum cholesterol and triglyceride levels. Although most of these changes were not clinically relevant, laboratory monitoring of patients with psoriasis treated with CyA is essential.

Creatinine↗

PubMed1978

Trauma from occlusion in monkeys.

Traumatic occlusion was induced in monkeys by insertion of bridges raising the bite. Both incisors and molars were used as abutments. Observation periods ranged from 7 to 24 months. No appreciable effect of the trauma on the apical proliferation of the crevicular epithelium was found. The results were the same in monkeys with or without marginal gingival inflammation before the trauma was induced.

Alveolar Process↗

PubMed1975

Effect of antiepileptic drugs on serum and salivary IgA.

IgA, IgG, and IgM concentrations were determined in sera from 100 patients under treatment for epilepsy for at least 6 months. Low IgA was found in 25% of adult sera and in an even larger percentage of sera from children. IgM concentrations were also low, whereas there was no significant difference in the IgG concentrations compared with normal sera. IgA values were normal in untreated patients but fell during treatment with phenytoin, in one patient from 1.5 to 0.2 mg/ml within 2 months. Low or undetectable salivary IgA was a common finding, especially in gingival hyperplasia.

Adolescent↗

PubMed2004

Cyclosporin A: a new drug in the field of canine dermatology.

In the last few years, there has been growing interest in the use of cyclosporin to treat canine skin diseases. Cyclosporin exhibits potent immunomodulating properties that reflect its ability to block the transcription of cytokines genes in activated T lymphocytes. Cyclosporin also inhibits a number of immune allergic reactions that occur after activation of mast cells, Langerhans cells, eosinophils and keratinocytes. In randomized controlled trials, cyclosporin has proven to be as effective as glucocorticoids for treatment of canine atopic dermatitis at the inducing dosage of 5 mg kg(-1). The drug has also proven beneficial for the treatment of perianal fistulas in dogs. Other potential applications are suggested from small pilot open trials using dogs affected with various immune-mediated dermatological diseases. The pharmacokinetic properties of cyclosporin are very similar in dogs and man, but its safety margin is much wider in dogs. Therefore, routine cyclosporin blood level monitoring does not appear necessary. Although in man renal impairment and hypertension are often seen, even at low doses, these effects are not observed in dogs. Adverse reactions consist mainly of transient emesis and diarrhoea occurring during the first days of treatment. Other adverse reactions, such as gingival hyperplasia, verruciform lesions and hypertrichosis, appear to be dose-dependent, and occur rarely at therapeutic doses. An increased susceptibility to infections has not been reported in dogs receiving this drug.

Administration, Oral↗

PubMed2006

Sweet's syndrome from an Indian perspective: a report of four cases and review of the literature.

BACKGROUND: Sweet's syndrome or acute febrile neutrophilic dermatosis is not frequently reported from India. Four patients fulfilling clinico-pathologic criteria for Sweet's syndrome seen during May-August 2002 prompted us to review reports on Indian patients from the indexed literature. METHODS: A PubMed and IndMed search for Sweet's syndrome revealed 11 reports appearing between 1985-2002 documenting 12 patients. The clinico-pathologic features, clinical course and treatment of all these 16 patients (including four new cases) were studied. RESULTS: The study comprised 12 females and four males between 35 days to 57 years of age. There were four children of < 12 years. Ulcerated, crusted lesions in one and typical, erythematous, tender, papulo-nodulo-plaques and targetoid lesions with pseudovesiculation were observed in 13 other patients particularly involving head, neck and upper limbs. Extracutaneous Sweet's syndrome manifesting as gingival hyperplasia and myositis was seen in one patient each. All had simultaneous onset of cutaneous lesions, fever, headache and malaise. Ocular involvement was observed in four patients only. Associated hematoligic disorders recorded were myelodysplasia in three, polycythemia vera in one and leukemia in three patients respectively. Two of these patients had treatment with all-transretinoic acid and low-dose cytosine-arbinoside before onset of Sweet's syndrome. One patient each had symptoms of upper respiratory tract infection and history of minor injury prior to cutaneous lesions. Another patient had pregnancy induced self-limiting Sweet's syndrome. Leukocytosis was present in 11 patients. Three of four new patients had positive tests for antistreptolysin-O and C-reactive protein. Characteristics histologic features were recorded in specimens of all patients. Eleven patients responded to systemic corticosteroids. Colchicin or potassium iodide were effective in one patient each. CONCLUSION: The overall clinico-pathologic and therapeutic spectrum of Sweet's syndrome in Indian patients does not appear to be different from its established picture.

Adult↗

PubMed1983

Mucolipidosis II. The clinical, radiological and biochemical features in three cases.

We report on the clinical, radiological and biochemical features of mucolipidosis II in three infants. One with subtle phenotypical findings died at 2 weeks of age without a specific diagnosis. A sibling who died at 2 years of age and another infant, presently 3.5 years of age manifest all the characteristic features of mucolipidosis II: extreme psychomotor delay and failure to thrive, coarse facial features, gingival hyperplasia, joint stiffness, inguinal hernia and skin induration. The corneae were normal and there was no mucopolysacchariduria. Radiologically, these infants show changes which are characteristic but not specific for mucolipidosis II. Cytologically, skin fibroblasts from these patients demonstrate the lysosomal inclusions typical of I-Cell Disease. Biochemically, cultured skin fibroblasts show deficient activity of arylsulphatase A and B and hexosaminidase A and B. These acid hydrolases were increased markedly in plasma and in the culture medium of the skin fibroblasts.

Cells, Cultured↗

PubMed1981

Mannosidosis: isolation and comparison of mannose-containing oligosaccharides from gingiva and urine.

Excessive gingival hyperplasia with storage of mannose-rich oligosaccharides appears to be a unique feature present in a 31-year-old mannosidosis patient. Fractionation and analysis of the gingiva established the presence of (Man)2GlcNAc (2.2 mumol/g), (Man)3GlcNAc (3.5 mumol/g), (Man)4GlcNAc (2.8 mumol/g) and higher oligomers (Man)5GlcNAc--(Man)8GlcNAc (0.5 mumol/g); (Man, mannose; GlcNAc, N-acetylglucosamine). Eight characteristic oligosaccharides were isolated from the patient's urine by thin-layer chromatography. The most abundant was (Man)2GlcNAc (161--558 mumol/l); decreasing amounts of higher homologues up to a dekasaccharide, (Man)9GlcNAc (1--4 mumol/l) were also present. In contrast to urine, in which the trisaccharide was predominant, tetrasaccharides and pentasaccharides were more abundant in gingiva.

Acetylglucosamine↗

PubMed2005

Skin disorders in patients transplanted in childhood.

Only few data are available on skin disorders in pediatric organ transplant recipients. In order to describe the whole range of dermatological diseases in a population of pediatric organ transplant recipients, we studied a group of 217 consecutive organ transplant recipients (168 kidney, 29 heart, 19 liver, one lung) aged <18 years at transplantation followed at a single center. A total of 193 patients showed at least one skin disorder; 149 had more than one skin disease. The most common skin infections were warts (24.4%), pityriasis versicolor (20.7%), folliculitis (12.9%), intertrigo (6.5%); the most common drug side effects were hypertrichosis (69.6%), steroid acne (39.6%), gingival hyperplasia (29%) and severe xerosis (20.7%). Two patients (0.9%) developed nonmelanoma skin cancer. Our study summarizes the main skin complications in patients transplanted in childhood and underlines the necessity of regular dermatologic surveillance of these patients.

Adolescent↗

PubMed1980

Study of the side effects of long-term anticonvulsant treatment.

In the present survey, we investigated the side effects of anticonvulsants in 248 epileptics who had been taking medicine for a long time. About half of the patients had been given anticonvulsant treatment for more than 11 years. The main results were as follows: Subjective symptoms: many kinds of gastrointestinal symptoms, general fatigability and sleepiness. slight pain in bones, joints or muscles and headache were found. Neurological symptoms: finger tremor at rest, diminished or decreased ankle reflex, and cerebellar symptoms such as ataxic gait, dysarthria, nystagmus and diplopia were found. Other clinical symptoms: gingival hyperplasia, hirsutism, dermatitis and edema were observed. Biochemical examinations: indicated that the total bilirubin was decreased in 4.4%, serum AL-P was elevated in 26.2%, the total serum cholesterol increased above 200 mg/dl in 17.7% and decreased below 150 mg/dl in 8.9%, and serum P and K were reduced in 31.5% and 2.4%, respectively. Hypocalcemia was found in only four cases (1.6%). Hematological examinations: serious disturbances were not found in hematopoietic functions, although prothrombin time was delayed in 18 of 40 patients examined.

Adolescent↗