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Oral verruciform hyperkeratotic lesions indicating the presence of plantar or palmoplantar keratodermas

  • Ioannis G. Koutlas
    Correspondence
    Corresponding author: Ioannis Koutlas
    Affiliations
    Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA
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  • Prokopios P. Argyris
    Affiliations
    Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA

    Department of Biochemistry, Molecular Biology and Biophysics, University of Minnesota, Minneapolis, MN, USA

    Masonic Cancer Center, University of Minnesota, Minneapolis, MN, USA

    Institute for Molecular Virology, University of Minnesota, Minneapolis, MN, USA

    Center for Genome Engineering, University of Minnesota, Minneapolis, MN, USA

    Howard Hughes Medical Institute, University of Minnesota, Minneapolis, MN, USA
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Published:April 21, 2022DOI:https://doi.org/10.1016/j.oooo.2022.04.044

      Objective

      Oral verruciform hyperkeratotic lesions (OVHLs) affecting the gingiva and palate are frequent in proliferative verrucous leukoplakia (PVL). Intraoral hyperkeratotic lesions can also be observed in epidermolytic keratodermas, albeit such association has received limited attention in oral and maxillofacial pathology. The authors report on 5 individuals whose plantar (PK) or palmoplantar keratodermas (PPKs) were confirmed after evaluation of gingival leukoplakic biopsies.

      Study Design

      Two women and 3 men, ages 18 to 64, presented with solitary or diffuse leukoplakias of the attached gingiva and hard palate, clinically interpreted as PVL. All individuals underwent diagnostic gingival and/or palatal biopsies.

      Results

      Microscopically, the lesions featured verruciform hyperparakeratosis, occasionally conspicuous hypergranulosis and acanthosis. In the spinous cell layer, numerous cells presented with vacuolated cytoplasm and paranuclear eosinophilic condensations that, infrequently, engulfed the nucleus. The histopathologic findings were interpreted as verrucous hyperkeratosis consistent with those described in epidermolytic PPKs. Further evaluation of the individuals for cutaneous lesions disclosed PK or PPKs in all 5 patients. Additionally, the men exhibited elbow and subungual hyperkeratoses. A family history of keratodermas was confirmed in all 3 male individuals.

      Conclusions

      Gingival and/or palatal OVHLs associated with PK and PPKs display pathognomonic histopathologic features and exhibit indolent biologic behavior. Therefore, any confusion with PVL should be avoided to prevent overtreatment.
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      References

        • Thompson LDR
        • Fitzpatrick SG
        • Müller S
        • et al.
        Proliferative verrucous leukoplakia: an expert consensus guideline for standardized assessment and reporting.
        Head Neck Pathol. 2021; 15: 572-587
        • Upadhyaya JD
        • Fitzpatrick SG
        • Cohen DM
        • et al.
        Inter-observer variability in the diagnosis of proliferative verrucous leukoplakia: clinical implications for oral and maxillofacial surgeon understanding: a collaborative pilot study.
        Head Neck Pathol. 2020; 14: 156-165
        • Bagan J
        • Scully C
        • Jimenez Y
        • Martorell M.
        Proliferative verrucous leukoplakia: a concise update.
        Oral Dis. 2010; 16: 328-332
        • Bagan JV
        • Jimenez Y
        • Sanchis JM
        • et al.
        Proliferative verrucous leukoplakia: high incidence of gingival squamous cell carcinoma.
        J Oral Pathol Med. 2003; 32: 379-382
        • Batsakis JG
        • Suarez P
        • el-Naggar AK.
        Proliferative verrucous leukoplakia and its related lesions.
        Oral Oncol. 1999; 35: 354-359
        • Young WG
        • Newcomb GM
        • Daley TJ.
        Focal palmoplantar and gingival hyperkeratosis syndrome: report of a family, with cytologic, ultrastructural, and histochemical findings.
        Oral Surg Oral Med Oral Pathol. 1982; 53: 473-482
        • Laskaris G
        • Vareltzidis A
        • Avgerinou G.
        Focal palmoplantar and oral mucosa hyperkeratosis syndrome: a report concerning five members of a family.
        Oral Surg Oral Med Oral Pathol. 1980; 50: 250-253
        • Kolde G
        • Hennies HC
        • Bethke G
        • Reichart PA.
        Focal palmoplantar and gingival keratosis: a distinct palmoplantar ectodermal dysplasia with epidermolytic alterations but lack of mutations in known keratins.
        J Am Acad Dermatol. 2005; 52: 403-409
        • Seehra J
        • Thakker N
        • Duxbury AJ
        • Sloan P
        • Ariyaratnam S.
        Focal palmoplantar and gingival hyperkeratosis: a case report.
        Dent Update. 2007; 34: 290-294
        • Gorlin RJ.
        Focal palmoplantar and marginal gingival hyperkeratosis—a syndrome.
        Birth Defects Orig Artic Ser. 1976; 12: 239-242
        • Guerra L
        • Castori M
        • Didona B
        • Castiglia D
        • Zambruno G.
        Hereditary palmoplantar keratodermas. Part I. Non-syndromic palmoplantar keratodermas: Classification, clinical and genetic features.
        J Eur Acad Dermatol Venereol. 2018; 32: 704-719
        • Patel S
        • Zirwas M
        • English JC 3rd
        Acquired palmoplantar keratoderma.
        Am J Clin Dermatol. 2007; 8: 1-11
        • Sakiyama T
        • Kubo A.
        Hereditary palmoplantar keratoderma “clinical and genetic differential diagnosis.
        J Dermatol. 2016; 43: 264-274
        • Christiano AM.
        Frontiers in keratodermas: pushing the envelope.
        Trends Genet. 1997; 13: 227-233
        • Kimyai-Asadi A
        • Kotcher LB
        • Jih MH.
        The molecular basis of hereditary palmoplantar keratodermas.
        J Am Acad Dermatol. 2002; 47: 327-346
        • Guerra L
        • Castori M
        • Didona B
        • Castiglia D
        • Zambruno G.
        Hereditary palmoplantar keratodermas. Part II: syndromic palmoplantar keratodermas—diagnostic algorithm and principles of therapy.
        J Eur Acad Dermatol Venereol. 2018; 32: 899-925
        • Leachman SA
        • Kaspar RL
        • Fleckman P
        • et al.
        Clinical and pathological features of pachyonychia congenita.
        J Investig Dermatol Symp Proc. 2005; 10: 3-17
        • Blaydon DC
        • Etheridge SL
        • Risk JM
        • et al.
        RHBDF2 mutations are associated with tylosis, a familial esophageal cancer syndrome.
        Am J Hum Genet. 2012; 90: 340-346
        • Alotaibi AK
        • Alotaibi MK
        • Alsaeed S
        • Alyahya A
        • Shuler CF.
        Olmsted syndrome with oral involvement, including premature teeth loss.
        Odontology. 2015; 103: 241-245
        • Fred HL
        • Gieser RG
        • Berry WR
        • Eiband JM.
        Keratosis palmaris et plantaris.
        Arch Intern Med. 1964; 113: 866-871
        • Argyris PP
        • Slama ZM
        • Nelson AC
        • Koutlas IG.
        Solitary oral epidermolytic acanthoma: case report of a rarely diagnosed entity.
        Oral Surg Oral Med Oral Pathol Oral Radiol. 2019; 128: e208-e213
        • Kerr AR
        • Lodi G.
        Management of oral potentially malignant disorders.
        Oral Dis. 2021; 27: 2008-2025