Complexity of dental management of a pediatric patient with lamellar ichthyosis: case report

Authors

DOI:

https://doi.org/10.33448/rsd-v12i4.41211

Keywords:

Ichthyosis, lamellar; Tooth, supernumerary; Pediatric dentistry.

Abstract

The purpose of this case report is to describe the complexity of dental management of a pediatric patient with lamellar ichthyosis. A 9-year-old male patient was referred by the pediatrician to the special needs’ clinic with a major complaint of "tooth in the roof of the mouth", according to the person in charge. The patient's mother reported that he was diagnosed with lamellar ichthyosis at birth and regularly followed up with a medical team, including a cardiologist, due to the presence of mild pulmonary valve stenosis. The physical examination revealed presence of skin peeling, everted lip, mouth breathing, cognitive deficit, and speech delay. The intraoral clinical examination showed mixed dentition, absence of caries lesions and presence of supernumerary teeth on the palate, between teeth 11 and 21, which was confirmed by radiographic examination. Although he was cleared by the pediatrician and cardiologist for the procedure under general anesthesia, this procedure in the hospital setting was interrupted at the last minute and contraindicated by the anesthesiologist, as it would be a high risky due to the patient's airway conditions. Thus, one month later, the patient's surgery was performed under local anesthesia on an outpatient basis under sedation and protective stabilization. The patient remains on follow-up and receiving preventive treatment. Thus, it is fundamental that there is a multidisciplinary approach of these patients to prevent possible complications resulting from dental procedures. In addition, regular dental monitoring is essential to avoid subjecting the patient to invasive procedures.

References

Aigner, C., Gajardo, A., & Matiello, M. (2008). Relato de caso: bebê arlequim ictiose congênita. Arquivos Catarineneses de Medicina, 37(4): 5356.

Akiyama, M. (2006). Pathomechanisms of harlequin ichthyosis and ABCA transporters in human diseases. Archives of Dermatology, 142:914-918.

Akiyama, M. (2011). Updated molecular genetics and pathogenesis of ichthiyoses. Nagoya Journal of Medical Science, 73: 79–90.

Crisóstomo, P. L., Brito, M. C., Pedrini, I. M., Sousa, D. C., Quintanilha, D. O., Santino, M. F. F., Fernandes, R. B. A., Villalba, R. C., & Sia, S. M. A. (2016). Ictiose Lamelar. Revista de Pediatria SOPERJ, 16(2):31-35.

Fitzpatrick, T. B., Freedberg, I. M. (1999) Fitzpatrick's dermatology in general medicine.

Fleckman, P., & DiGiovanna, J. J. (2008). Fitzpatrick’s Dermatology in General Medicin.; (7th ed.).

Gulasi, S. (2016). Congenital Ichthyosis: A Case Treated Successfully with Acitretin. Iranian Journal of Pediatrics, 26(5): e2442.

Hosal, B. M., Abbasoglu, O. E., & Gursel, E. (1999). Surgical treatment of cicatricial ectropion in lameller ichthyosis. Orbit,19: 37–40.

Lefèvre, C., Bouadjar, B., Ferrand, V., Tadini, G., Mégarbané, A., Lathrop, M., Prud'homme, Jean-François., & Fischer, J. (2006). Mutations in a new cytochrome P450 gene in lamellar ichthyosis type 3. Human Molecular Genetics, 15(5):767–776.

Matsuno, C. A., Santana, L. O. B., Simis, D .R .C., Barbo, M. L. P., & Vieira, M. W. (2014). Lamellar ichthyosis: a case report. Revista da Faculdade de Ciências Médicas de Sorocaba, 16(3):146–148.

Neema, S., Mukherjee, S., Vasudevan, B., Verma, R., Moorchung, N., & Chatterjee, M. (2015). Vitamin D Deficiency After Oral Retinoid Therapy for Ichthyosis. Pediatric Dermatology, 32(4):e151-5.

Oji, V., Tadini, G., Akiyama, M., Blanchet Bardon, C., Bodemer, C., Bourrat, E., et al. (2010). Revised nomenclature and classification of inherited ichthyoses: results of the First Ichthyosis Consensus Conference in Soreze 2009. Journal of the American Academy of Dermatology, 63: 607–641.

Orfanos, C .E., Zouboulis, C .C., Almond-Roesler, B., & Geilen, C. C. (1997). Current use and future potential role of retinoids in dermatology. Drugs, 53:358–88.

Pereira, A. S., Shitsuka, D. M., Parreira, F. J., & Shitsuka, R. (2018). Metodologia da pesquisa científica. UFSM. https://repositorio.ufsm.br/bitstream/handle/1/15824/Lic_Computacao_Metodologia-Pesquisa-Cientifica. pdf

Pranitha, V., Thimma Reddy, B. V, Daneswari, V., & Deshmukh, S .N. (2014). Lamellar Icthyosis - A case Report. Journal of Clinical and Diagnostic Research: JCDR, 8(11): ZD01-2.

Prasad, R. S., Pejaver, R. K., Hassan, A., al Dusari, S., & Wooldridge, M. A. (1994). Management and follow-up of harlequin siblings. British Journal of Dermatology, 130:650-653.

Ramar, K., Annamalai, S., Hariharavel, V. P., Aravindhan, R., Ganesh, C., & Ieshwaryah, K. (s.d.). Oral manifestation of autosomal recessive congenital ichthyosis in a 2-year-old patient. Case Reports in Dentistry, 2014:483293.

Reis, G. E. S., Marchetti, G., Casatti, B. L. V. M., Sarot, J. R., Menezes, J. S. N .B., & Pupo, Y. M. (2022). Oral Manifestations and Dental Approach in Pediatric Patient with Lamellar Congenital Ichthyosis: 36-Month Follow-up. Revista Científica do CRO-RJ, 7(2).

Singh, A. J., & Atkinson, P. L. (2005). Ocular manifestations of congenital lamellar ichthyosis. European Journal of Ophthalmology, 15(1):118-122.

Takeichi, T., & Akiyama, M. (2016). Inherited ichthyosis: Non-syndromic forms. The Journal of Dermatology, 43(3):242-51.

Taieb, A., & Labreze C. (2002). Collodion baby: whats new. European Academy of Dermatology and Venereology, 16:4367.

Tewari, N., Mathur, V. P., Tamchos, R., & Rahul, M. (2020). Oral manifestations of lamellar ichthyosis in association with rickets. BMJ Case Reports, 13(7):e235008.

Uthoff, D., Gorney, M., & Teichmann, C. (1994). Cicatricial ectropion in ichthyosis: a novel approach to treatment. Ophthalmic Plastic & Reconstructive Surgery, 10:92-5.

Yang, J. M., Ahn, K. S., Cho, M. O., Yoneda, K., Lee, C. H., Lee, J. H., Lee, E. S., Candi, E., Melino, G., Ahvazi, B., & Steinert, P. M. (2001). Novel mutations of the transglutaminase 1 gene in lamellar ichthyosis. Journal of Investigative Dermatology, 117(2):214-8.

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Published

16/04/2023

How to Cite

RODRIGUES, G. F. .; LOSSO, A. R. .; MORAES, R. dos R. .; DUARTE, M. L. .; MACEDO, R. G. .; CASTRO, G. F. B. de A. Complexity of dental management of a pediatric patient with lamellar ichthyosis: case report . Research, Society and Development, [S. l.], v. 12, n. 4, p. e23912441211, 2023. DOI: 10.33448/rsd-v12i4.41211. Disponível em: https://rsdjournal.org/index.php/rsd/article/view/41211. Acesso em: 23 nov. 2024.

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Section

Health Sciences