Review of linear epidermal nevus with oral mucosal involvement--series of five new cases.

INTRODUCTION: Linear epidermal nevi are sporadic hamartomatous alterations of the epidermis and superficial dermis that clinically appear as verrucous papules and plaques distributed in a linear pattern following Blaschko's lines. Their extent varies from unilateral involvement (nevus unius lateris) to extensive bilateral involvement (ichthyosis hystrix). Oral mucosal lesions have rarely been described. AIMS: We review the literature, focusing on the rare intraoral manifestations of linear epidermal nevus. CASE SERIES: We present a series of five new cases with oral mucosal involvement. Four cases had associated cutaneous lesions and one case had oral lesions exclusively. Histopathologic evaluation of lesional tissue in four cases showed hyperkeratosis, acanthosis, epithelial hyperplasia, and papillomatosis. Dental abnormalities, consisting of enamel hypoplasia and congenitally missing teeth, were noted in one patient adjacent to the oral lesions.

Caliber-persistent labial artery. A common vascular anomaly.

Sixteen cases of caliber-persistent labial artery of the lips have been reported to date in the English literature. Six of these were clinically misdiagnosed as squamous cell carcinoma and treated with wedge resection. To date, we have seen 187 cases clinically and an additional 23 cases through our surgical oral pathology services. Careful clinical observation usually reveals a soft linear or papular bluish elevation above the labial mucosal surface. The unique feature is pulsation--not simply pulsation toward and away from the observer, which can be caused by an underlying artery, but lateral pulsation, which only an artery can exhibit. All but 2 of our 187 clinical cases were asymptomatic. To the best of our knowledge, this is the first report of caliber-persistent labial artery of the upper lip. The upper:lower lip ratio for the clinical cases was almost 2:1. Three times as many lower lip as upper lip lesions were biopsied. Males and females were almost equally affected (clinical cases, 76:86; histopathologic cases, 9:13). Although a vascular term (artery, hemangioma, phlebolith, varix, vascular malformation) was used on the biopsy form in one half of the clinical differential diagnoses, none of the clinical histories mentioned pulsation. In contrast to the cases of Miko et al. in 1980 and 1983, none of our cases manifested itself as an ulcer, nor was carcinoma ever mentioned in the clinical differential diagnosis. The purpose of this article is to familiarize clinicians and pathologists with the clinical and histopathologic features of this seldom reported but common vascular anomaly. Clinicians should carefully look for lateral pulsation in lip mucosal papules so as to avoid unnecessary surgery and intraoperative arterial bleeding. Pathologists should recognize that a relatively large-caliber superficial artery in a lip biopsy may not be an incidental finding but rather the clinical lesion that was biopsied.

Evaluation of serum and tissue levels of alpha-tocopherol.

This study was designed to test the effect of supplementation of several antioxidants, including alpha-tocopherol, on the clinical reduction of premalignant oral lesions. Samples of oral mucosa and serum were taken from baseline to 9 months of supplementation from patients with premalignant oral lesions and analyzed for alpha-tocopherol by HPLC. Statistical increases in both serum and tissue alpha-tocopherol were found after supplementation. There was no statistical relationship between alpha-tocopherol and beta-carotene levels.

Salivary gland duct involvement in oral epithelial dysplasia and squamous cell carcinoma.

The clinical implications and prognostic significance of oral dysplastic or cancerous epithelium involving salivary gland ducts have not been previously investigated. Screened routine tissue sections of 1216 cases of oral epithelial dysplasias and squamous cell carcinomas revealed 26 examples (2.14%) that exhibited unequivocal ductal involvement. Ductal involvement was more likely to occur in floor of mouth lesions and in lesions exhibiting severe dysplasia or carcinoma in situ. Clinical follow-up on 23 cases showed that the recurrence rate of the preinvasive lesions that exhibited ductal involvement was equal to that of the squamous cell carcinomas. The depth of ductal dysplasia did not correlate with recurrence rate. These results suggest that the involvement of salivary gland ducts by oral epithelial dysplasias and carcinomas in situ is an uncommon but significant finding. Surgical stripping or ablation of such lesions should extend at least 3 mm below the surface to ensure eradication of these reservoirs of dysplastic cells.

Use of antioxidant supplements in the treatment of human oral leukoplakia.

An increasing public awareness of antioxidants may prompt a patient's request to be treated without surgery if a leukoplakic lesion is discovered. However, surgical excision remains the treatment of choice for oral leukoplakia. The use of antioxidant supplements has shown some promise, but the predictability of success remains uncertain and long-term results are unavailable. Before the decision to use any antioxidant is made, it is critical to obtain a histopathologic diagnosis of the lesion. When dealing with a lesion diagnosed as hyperkeratosis, it may be appropriate to choose an antioxidant that may take some time for clinical improvement to occur. However, as the grade of epithelial dysplasia becomes more severe, consideration must be given to the possibility of malignant transformation during antioxidant treatment. We do not recommend the use of antioxidant supplements in the treatment of any carcinoma. The therapeutic use of antioxidant supplements outside of clinical trials conducted at academic medical centers should be done with considerable caution by practitioners in private practice. It should be emphasized that in these clinical trial patients were seen at frequent intervals to monitor their progress and to intervene if there was a noticeable deterioration in the clinical appearance of the lesion. In spite of the uncertainty with respect to antioxidant treatment, there are circumstances in which it should be considered. Recurrence after surgical excision when there is little reason to believe that a second surgical excision would be any more successful is an ideal candidate. Also, patients with widespread leukoplakia that involves a large area of the oral mucosa might be suitable for treatment with antioxidants, as well as patients who have extensive medical problems that make them surgical risks. The choice of which antioxidant(s) to use is complex because thus far there is no combination that is superior to the others. Beta-carotene with ascorbic acid or alpha-tocopherol is attractive because of a lack of side effects, but the range in reported values for lesion improvement has been broad and the clinical improvement typically takes several months. Clinical response with 13-cRA is faster but requires baseline and periodic serologic testing, as well as close monitoring for side effects. In those circumstances in which time is an important consideration, 13-cRA might be useful because clinical improvement can be evaluated within a matter of weeks as compared with beta-carotene. The group from M.D. Anderson Hospital has shown the value of an induction dose of 13-cRA that is followed by a lower maintenance dose. Unfortunately, the problem of recurrence after discontinuation of 13-cRA is quite common. One aspect that has not been evaluated is the combination of conventional surgical excision and the administration of postoperative antioxidants. This would have the obvious advantage of conventional treatment of surgery together with the possible protective effect of the antioxidants. Although this is an attractive hypothesis, we do not know of any studies that have proven this to be beneficial.

Lupus erythematosus-like oral mucosal and skin lesions in a carrier of chronic granulomatous disease. Chronic granulomatous disease carrier genodermatosis.

We present a case of a young female patient who for 8 years was believed to have discoid lupus erythematosus of the skin and oral mucosae. Only after her infant son had a near-fatal pulmonary infection was the diagnosis of chronic granulomatous disease made and her lupus erythematosus-like mucocutaneous lesions recognized as manifestations of her carrier status for chronic granulomatous disease. The purpose of this report is to raise awareness of and better characterize the mucocutaneous manifestations of carriers of chronic granulomatous disease. Early identification of carriers permits genetic counseling and prenatal diagnosis and forewarns pediatricians so that they can provide better care for affected infants.

A clinical trial of antioxidant supplements in the treatment of oral leukoplakia.

Seventy-nine patients with oral leukoplakia that was histologically verified as either hyperkeratosis or epithelial dysplasia with hyperkeratosis were enrolled in an antioxidant supplementation program for the treatment of the oral lesions. The patients received 30 mg of beta-carotene, 1000 mg of ascorbic acid, and 800 IU of alpha-tocopherol per day for 9 months. Clinical improvement of the oral lesion was noted in 55.7% of the patients and was more likely to occur in patients who reduced their use of alcohol or tobacco (p = 0.0056). Although risk-factor reduction was important, approximately half of the patients who did not alter their exposure to either alcohol or tobacco showed clinical improvement. The antioxidant supplementation significantly increased serum and tissue levels of beta-carotene, ascorbic acid, and alpha-tocopherol, but these changes did not correlate strongly with clinical improvement.

Evaluation of serum and tissue levels of beta-carotene.

Interest in beta-carotene (BC) has increased as studies show that low dietary or serum BC is associated with increased risk of cancer. Patients with oral epithelial dysplasia had serum and oral mucosa punch biopsy samples taken before supplementation of 30 mg/day of beta-carotene and after 6 and 9 months of supplementation. BC was analyzed for 28 patients by high-performance liquid chromatography. At baseline, serum BC and gender accounted for 48% of tissue BC variance: Tissue BC = -0.13 + 0.08(Serum BC) + 1.21(Sex) with sex as male = 0 or female = 1. Following supplementation, serum BC had an exponential relationship to tissue BC which accounted for 52% of tissue variance: Tissue BC = 1.15 + 5.7 x 10(-5)(Serum BC)2 + 3.91(Sex). Women had higher mean concentrations of serum and oral mucosal BC before and during supplementation. The need for oral mucosal sampling may be eliminated by the correlation between serum and tissue BC.

Clinical diagnosis of caliber-persistent labial artery of the lower lip.

Only 14 cases of caliber-persistent labial artery of the lower lip have been reported to date. Six of these were misdiagnosed and treated as squamous cell carcinoma, another as a mucocele. The correct diagnosis emerged only after the wedge resections were examined histopathologically. We report the first cases of caliber-persistent labial artery to be diagnosed clinically since the original description of the condition by Howell and Freeman in 1973. Our first case was a nonpulsatile hard, linear, "gooseneck lamp" submucosal nodule of the lower lip. On the basis of an initial misdiagnosis of sclerosing sialadenitis, a biopsy was attempted. Brisk pulsatile bleeding proved the lesion to be an artery, and the superficial location and large diameter of the vessel lead to the clinical diagnosis of caliber-persistent labial artery. The "gooseneck lamp" hardening is typical of Monckeberg's arteriosclerosis. The second case was a pulsatile blue linear submucosal nodule of the lower lip. The clinical diagnosis of caliber-persistent labial artery was confirmed when angiography showed the lesion to be an abnormally dilated labial artery. Both cases were successfully ligated with no complications at 16 and 10 months after surgery.

A review of the use of antioxidant supplements in the treatment of human oral leukoplakia.

Over the past twenty years, research into the role of antioxidants in the prevention of cancer has increased dramatically. The use of antioxidant supplements to treat oral leukoplakia has gained acceptance due to the success demonstrated in several clinical trials. This review discusses the role of antioxidants in the development of cancer and their possible use in the treatment of oral leukoplakia.

Oral pathology in Canada: a survey.

The Canadian Academy of Oral Pathology conducted a survey of its active members to obtain information on their demographics, professional activities and university affiliations, and to assess Canada's present and future oral pathology needs. Data were also collected on Canadian oral pathology diagnostic biopsy services. This paper reports the salient findings of the survey, and discusses their implications for the specialty of oral pathology as well as for the Canadian dental community.

Diseases of the salivary glands: a review.

The normal function of major salivary glands as well as thousands of minor ones is an important determinant of oral health. Many diseases of these glands are common, yet not often appreciated by the dentist or his staff. This paper reviews common and some less common diseases of salivary glands, including developmental, hypertrophic, reactive, autoimmune and neoplastic conditions.

Oral lichenoid dysplasia: a clinicopathologic analysis.

Three women had a diagnosis of oral lichen planus (OLP), which was made on the basis of clinical and histologic features. All three had persistent burning pain associated with large mucosal lesions. Changes in the color (red, red and white, white), configuration, and severity of the lesions were unpredictable and did not correlate well with topical corticosteroid therapy. Only one patient used tobacco (cigarettes)--this patient had recurrent oral candidiasis and was receiving multiple medications. One of the two nonsmokers was a denture wearer with a single episode of candidiasis. After 63, 32, and 56 (mean 50) months, carcinoma developed in all three. In retrospect, the initial biopsy specimens of two patients exhibited lichenoid dysplasia, whereas that of the third showed only lichenoid mucositis. Although speckled erythroplakia was the earliest clinical sign of a classic, nonregressing premalignant lesion, it already signaled the presence of invasive carcinoma. Some early epithelial dysplasias appear to have a robust inflammatory/immunologic response to the antigenically (but as yet not histologically) altered dysplastic epithelium and a high probability of at least temporary resolution. Mucosal erythema of obscure origin displaying spontaneous, usually temporary, partial to complete regression may be common to both purely inflammatory conditions like lichen planus and early epithelial dysplasia. We contend that some, if not most, cases of apparent malignant transformation of OLP likely represent red and white lesions that were dysplastic from their inception but that mimic OLP both clinically and histologically.