Laugier–Hunziker Syndrome Presenting with Metachronous Melanoacanthomas

  • Hattan Zaki
  • Amarpreet Sabharwal
  • Jill Kramer
  • Alfredo Aguirre
Case Report

Abstract

Laugier–Hunziker syndrome (LHS, also termed idiopathic lenticular mucocutaneous hyperpigmentation) is an unusual condition characterized by progressive pigmentation of the mucous membranes. LHS displays a benign course and is not associated with malignancy. Here we present a case of LHS with a 7-year follow-up. We document metachronous oral melanoacanthomas in this individual. In addition, we found that the oral melanotic macules in this patient waxed and waned in a cyclical manner. To our knowledge, this is the first report of these findings in the context of LHS. Finally, we provide an overview of other conditions that can present with mucosal hyperpigmentation. It is critical to distinguish LHS from other conditions characterized by mucosal pigmentation in order to facilitate optimal patient care.

Keywords

Laugier–Hunziker syndrome Oral melanotic macule Melanoacanthoma Melanin Hyperpigmentation 

Notes

Compliance with Ethical Standards

Conflict of interest

All authors declare that they have no conflicts of interest.

Ethical Approval

This article does not contain any studies with human participants or animals performed by any of the authors.

References

  1. 1.
    Laugier P, Hunziker N. Essential lenticular melanic pigmentation of the lip and cheek mucosa. Arch Belg Dermatol Syphiligr. 1970;26(3):391–9.PubMedGoogle Scholar
  2. 2.
    Baran R. Longitudinal melanotic streaks as a clue to Laugier–Hunziker syndrome. Arch Dermatol. 1979;115(12):1448–9.CrossRefPubMedGoogle Scholar
  3. 3.
    Kanwar AJ, Kaur S, Kaur C, Thami GP. Laugier–Hunziker syndrome. J Dermatol. 2001;28(1):54–7.CrossRefPubMedGoogle Scholar
  4. 4.
    Mignogna M, Muzio LL, Ruoppo E, Errico M, Amato M, Satriano R. Oral manifestations of idiopathic lenticular mucocutaneous pigmentation (Laugier–Hunziker syndrome): a clinical, histopathological and ultrastructural review of 12 cases. Oral diseases. 1999;5(1):80–6.CrossRefPubMedGoogle Scholar
  5. 5.
    Siponen M, Salo T. Idiopathic lenticular mucocutaneous pigmentation (Laugier–Hunziker syndrome): a report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96(3):288–92.CrossRefPubMedGoogle Scholar
  6. 6.
    Wang WM, Wang X, Duan N, Jiang HL, Huang XF. Laugier–Hunziker syndrome: a report of three cases and literature review. Int J Oral Sci. 2012;4(4):226–30.CrossRefPubMedPubMedCentralGoogle Scholar
  7. 7.
    Makhoul EN, Ayoub NM, Helou JF, Abadjian GA. Familial Laugier–Hunziker syndrome. J Am Acad Dermatol. 2003;49(2):143–5.CrossRefGoogle Scholar
  8. 8.
    Bhoyrul B, Paulus J. Macular pigmentation complicating irritant contact dermatitis and viral warts in Laugier–Hunziker syndrome. Clin Exp Dermatol. 2016;41(3):294–6.CrossRefPubMedGoogle Scholar
  9. 9.
    Cusick EH, Marghoob AA, Braun RP. Laugier–Hunziker syndrome: a case of asymptomatic mucosal and acral hyperpigmentation. Dermatol Pract Concept. 2017;7(2):27–30.CrossRefPubMedPubMedCentralGoogle Scholar
  10. 10.
    Mahmood T, Menter A. The Laugier–Hunziker syndrome. Proc (Bayl Univ Med Cent). 2015;28(1):41–2.CrossRefGoogle Scholar
  11. 11.
    Sendagorta E, Feito M, Ramirez P, Gonzalez-Beato M, Saida T, Pizarro A. Dermoscopic findings and histological correlation of the acral volar pigmented maculae in Laugier–Hunziker syndrome. J Dermatol. 2010;37(11):980–4.CrossRefPubMedGoogle Scholar
  12. 12.
    Gerbig AW, Hunziker T. Idiopathic lenticular mucocutaneous pigmentation or Laugier–Hunziker syndrome with atypical features. Arch Dermatol. 1996;132(7):844–5.CrossRefPubMedGoogle Scholar
  13. 13.
    Nikitakis NG, Koumaki D. Laugier–Hunziker syndrome: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2013;116(1):e52–8.CrossRefGoogle Scholar
  14. 14.
    Yago K, Tanaka Y, Asanami S. Laugier–Hunziker-Baran syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;106(2):e20–5.CrossRefPubMedGoogle Scholar
  15. 15.
    Higham P, Alawi F, Stoopler ET. Medical management update: Peutz Jeghers syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2010;109(1):5–11.CrossRefPubMedGoogle Scholar
  16. 16.
    Brandao Neto RA, de Carvalho JF. Diagnosis and classification of Addison’s disease (autoimmune adrenalitis). Autoimmun Rev. 2014;13(4–5):408–11.CrossRefPubMedGoogle Scholar
  17. 17.
    Adel Kauzman B, Pavone M, Blanas N, Bradley G. Pigmented lesions of the oral cavity: review, differential diagnosis, and case presentations. J Can Dent Assoc. 2004;70(10):682–3.PubMedGoogle Scholar
  18. 18.
    Eisen D. Disorders of pigmentation in the oral cavity. Clin Dermatol. 2000;18(5):579–87.CrossRefPubMedGoogle Scholar
  19. 19.
    Lenane P, Powell F. Oral pigmentation. J Eur Acad Dermatol Venereol. 2000;14(6):448–65.CrossRefPubMedGoogle Scholar
  20. 20.
    Müller S. Melanin-associated pigmented lesions of the oral mucosa: presentation, differential diagnosis, and treatment. Dermatol Ther. 2010;23(3):220–9.CrossRefPubMedGoogle Scholar
  21. 21.
    Lampe A, Hampton P, Woodford-Richens K, Tomlinson I, Lawrence C, Douglas F. Laugier–Hunziker syndrome: an important differential diagnosis for Peutz-Jeghers syndrome. J Med Genet. 2003;40(6):e77–e77.CrossRefPubMedPubMedCentralGoogle Scholar
  22. 22.
    Amir E, Gorsky M, Buchner A, Sarnat H, Gat H. Physiologic pigmentation of the oral mucosa in Israeli children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1991;71(3):396–8.CrossRefGoogle Scholar
  23. 23.
    Gondak RO, da Silva-Jorge R, Jorge J, Lopes MA, Vargas PA. Oral pigmented lesions: clinicopathologic features and review of the literature. Med Oral Patol Oral Cir Bucal. 2012;17(6):e919–24.CrossRefPubMedPubMedCentralGoogle Scholar
  24. 24.
    Neville B, Damm D, Chi A, Allen C. Physical and chemical injuries. In: Neville B, Damm D, Chi A, Allen C, editors. Oral and maxillofacial pathology. 4th ed. St. Louis: Elsevier Health Sciences; 2016. pp. 289–90.Google Scholar
  25. 25.
    Muller S. Melanin-associated pigmented lesions of the oral mucosa: presentation, differential diagnosis, and treatment. Dermatol Ther. 2010;23(3):220–9.CrossRefPubMedGoogle Scholar
  26. 26.
    Abdollahi M, Radfar M. A review of drug-induced oral reactions. J Contemp Dent Pract. 2003;4(1):10–31.PubMedGoogle Scholar
  27. 27.
    Lerman MA, Karimbux N, Guze KA, Woo S-B. Pigmentation of the hard palate. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009;107(1):8–12.CrossRefPubMedGoogle Scholar
  28. 28.
    Krause W. Drug-induced hyperpigemntation: a systematic review. J Dtsch Dermatol Ges. 2013;11(7):644–51.PubMedGoogle Scholar
  29. 29.
    Wilder EG, Frieder J, Sulhan S, Michel P, Cizenski JD, Wright JM, et al. Spectrum of orocutaneous disease associations: genodermatoses and inflammatory conditions. J Am Acad Dermatol. 2017;77(5):809–30.CrossRefPubMedGoogle Scholar
  30. 30.
    Hemminki A, Markie D, Tomlinson I, Avizienyte E, Roth S, Loukola A, et al. A serine/threonine kinase gene defective in Peutz–Jeghers syndrome. Nature. 1998;391(6663):184–7.CrossRefPubMedGoogle Scholar
  31. 31.
    Hemminki A, Tomlinson I, Markie D, Jarvinen H, Sistonen P, Bjorkqvist AM, et al. Localization of a susceptibility locus for Peutz–Jeghers syndrome to 19p using comparative genomic hybridization and targeted linkage analysis. Nat Genet. 1997;15(1):87–90.CrossRefPubMedGoogle Scholar
  32. 32.
    Pereira CM, Coletta RD, Jorge J, Lopes MA. Peutz-Jeghers syndrome in a 14-year-old boy: case report and review of the literature. Int J Paediatr Dent. 2005;15(3):224–8.CrossRefPubMedGoogle Scholar
  33. 33.
    Duong BT, Winship I. The role of STK 11 gene testing in individuals with oral pigmentation. Australas J Dermatol. 2017;58(2):135–8.CrossRefPubMedGoogle Scholar
  34. 34.
    Rohilla K, Ramesh V, Balamurali P, Singh N. Oral Melanoacanthoma of a rare intraoral site: case report and review of literature. Int J Clin Pediatr Dentist. 2013;6(1):40.CrossRefGoogle Scholar
  35. 35.
    Fatahzadeh M, Sirois DA. Multiple intraoral melanoacanthomas: a case report with unusual findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94(1):54–6.CrossRefPubMedGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  1. 1.Oral Basic and Clinical Sciences Department, Faculty of DentistryTaibah UniversityMadinahSaudi Arabia
  2. 2.Department of Periodontics and Endodontics, School of Dental MedicineUniversity at Buffalo, The State University of New YorkBuffaloUSA
  3. 3.Department of Oral Biology, School of Dental MedicineUniversity at Buffalo, The State University of New YorkBuffaloUSA
  4. 4.Department of Oral Diagnostic Sciences, School of Dental MedicineUniversity at Buffalo, The State University of New YorkBuffaloUSA

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