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Focal palmoplantar and gingival keratosis - A rare genodermatoses: Case report.

ABSTRACT: Focal palmoplantar and gingival keratosis syndrome is a rare dominant inherited disease with an early onset in life. Clinically, the condition is characterized by pressure related thickening of the epidermis of the palms and soles, usually accompanied by pain and different levels of skin involvement and thickness between patients. Recently, we observed a 38-year-old woman with multiple non-removable, painless white plaques of variable size and thickness on the attached gingiva and a white plaque widespread across the hard palate. By further questioning, the patient comments that she has thick yellowish focal plaques in both soles of her feet. Histopathological analysis revealed a hyperplastic and hyperorthokeratinized stratified squamous epithelium with basal hyperplasia. The spinous, granular and stratum corneum showed dispersed basophilic keratohyalin granules. At higher magnification, the keratinocytes contained paranuclear bodies, seen as round eosinophilic condensation that indented the nuclei. Based on these findings the final diagnosis was rendered as focal palmoplantar and gingival keratosis. Key words:Genodermatoses, white plaque, dyskeratosis, gingiva.

SUBMITTER: Gerber-Mora R

PROVIDER: S-EPMC7680573 | biostudies-literature | 2020 Nov

REPOSITORIES: biostudies-literature

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Focal palmoplantar and gingival keratosis - A rare genodermatoses: Case report.

Gerber-Mora Roberto R Jajam-Maturana Martín M Fernández-Moraga Alejandra A Martinez-Flores Rene R

Journal of clinical and experimental dentistry 20201101 11

Focal palmoplantar and gingival keratosis syndrome is a rare dominant inherited disease with an early onset in life. Clinically, the condition is characterized by pressure related thickening of the epidermis of the palms and soles, usually accompanied by pain and different levels of skin involvement and thickness between patients. Recently, we observed a 38-year-old woman with multiple non-removable, painless white plaques of variable size and thickness on the attached gingiva and a white plaque ...[more]

PMID: 33262878

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Project description:BackgroundArsenical keratosis is a precancerous dermatosis which could be induced by long-term exposure to arsenic poisoning. Arsenic is often added to traditional Chinese medicine in a non-compliant manner to increase the effectiveness of psoriasis treatment, which is often the main cause of arsenic poisoning in Chinese patients with psoriasis.ObjectivesWe performed a systemic review of arsenic keratosis during the past 32 years to better understand the sources, treatment, and prognosis of arsenic keratosis in China.MethodsWe searched Medline/PubMed, Embase, CNKI, and Wanfang databases for research studies published between 1992 and 2024. A total of 64 papers with 78 individual Chinese of arsenical keratosis were included in this analysis.ResultsOf the patients included in the analysis, 92.21% of arsenic poisoning was due to iatrogenic factors: Chinese traditional medicine. Seventy-six patients (98.70%) had skin manifestation of hyperkeratotic papules and plaques, 68 patients (88.31%) had hyperpigmentation, 43 cases (55.84%) had hypopigmentation, and only 4 had a clear indication of Mees' lines in nails. A total of 52.63% of patients presented with tumors, including squamous cell carcinoma, Bowen's disease, and basal cell carcinoma. For patients with tumors, 20 opted for surgery, 6 for radiotherapy, and 3 for PDT. All patients with only cutaneous tumors are currently well-controlled. Death occurred in one patient with metastatic squamous cell carcinoma. Keratinizing papules improved significantly in 70.59% of patients treated with Acitretin Capsules.ConclusionsIn this study, arsenic sources in Chinese patients were mainly from traditional Chinese medicine, and there were no reports of exposure to water sources or occupational sources in the past 32 years. Most of the patients showed keratinizing papules and pigmentation, and more than 1/2 of the patients showed skin tumors, mainly squamous cell carcinoma. The treatments of tumors are mainly surgical treatment, PDT and radiotherapy can also be selected. The improvement in keratinizing rash was greater than 70% with acitretin capsules. Patients with this disease should be regularly followed up for early detection and timely treatment of potential malignant tumors.

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Focal palmoplantar and gingival keratosis - A rare genodermatoses: Case report.

Publications

Focal palmoplantar and gingival keratosis - A rare genodermatoses: Case report.

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