Congenital atresia of the submandibular gland duct: report of two clinical cases with spontaneous regression and literature review
Article Sidebar
Main Article Content
Background: Congenital atresia of the submandibular gland duct is a rare condition, originated during the embryogenic process, characterized by swelling in the region of the oral floor. A few cases of congenital atresia of the submandibular gland duct have appeared in the literature and only one reports no surgical intervention to date.
Case series: In the present study reports two cases of babies with clinical findings demonstrating swelling well-defined in the region of the oral floor, slightly translucent bluish color, painless to palpation, without finding stones or salivary drainage and apparently with no perforation of the submandibular gland duct. Both patients were kept under observation, later presenting spontaneous regression.
Conclusion: This study highlights the importance of conducting a thorough clinical examination in addition to showing that the use of complementary exams may not be essential.
Lara Oliveira Freitas, Universidade Federal dos Vales do Jequitinhonha e Mucuri
Universidade Federal dos Vales do Jequitinhonha e Mucuri, Faculty of Biological and Health Sciences, Department of Dentistry, Diamantina, Minas Gerais, Brazil.
Priscila Seixas Mourão, Universidade Federal dos Vales do Jequitinhonha e Mucuri
Universidade Federal dos Vales do Jequitinhonha e Mucuri, Faculty of Biological and Health Sciences, Department of Dentistry, Diamantina, Minas Gerais, Brazil.
Moisés Willian Aparecido Gonçalves, Universidade Federal dos Vales do Jequitinhonha e Mucuri
Universidade Federal dos Vales do Jequitinhonha e Mucuri, Faculty of Biological and Health Sciences, Department of Dentistry, Diamantina, Minas Gerais, Brazil.
Maria Letícia Ramos-Jorge, Universidade Federal dos Vales do Jequitinhonha e Mucuri
Universidade Federal dos Vales do Jequitinhonha e Mucuri, Faculty of Biological and Health Sciences, Department of Dentistry, Diamantina, Minas Gerais, Brazil.
Ana Terezinha Marques Mesquita , Universidade Federal dos Vales do Jequitinhonha e Mucuri
Universidade Federal dos Vales do Jequitinhonha e Mucuri, Faculty of Biological and Health Sciences, Department of Dentistry, Diamantina, Minas Gerais, Brazil.
Gibson MHL. The prenatal human submandibular gland: a histological, histochemical and ultrastructural study. Anat Anz. 1983;153:91.
Garcia-Garcia JD, Merida-Velasco JA, Barranco-Zafra RJ, Espin-Ferra J, Sanchez-Montesinos I. Development of ductus submandibularis in the human submandibular gland. Eur Arch Biol. 1991;102:1-7.
Amin MA, Bailey BMW. Congenital atresia of the orifice of the submandibular duct: a report of 2 cases and review. Br J Oral Maxillofac Surg. 2001;39:480-2. 2001. https://doi.org/10.1054/bjom.2001.0672 DOI: https://doi.org/10.1054/bjom.2001.0672
Walker P. Imperforate submandibular duct. Otolaryngol Head Neck Surg. 2005;132:653-4. https://doi.org/10.1016%2Fj.otohns.2004.09.023 DOI: https://doi.org/10.1016/j.otohns.2004.09.023
Mandel L, Alfi D. Diagnostic imaging for submandibular duct atresia: literature review and case report. J Oral Maxillofac Surg. 2012;70:2819-22. http://dx.doi.org/10.1016/j.joms.2012.02.032 DOI: https://doi.org/10.1016/j.joms.2012.02.032
Hoffrichter MS, Obeid G, Soliday JT. Bilateral submandibular duct atresia: case report. J Oral Maxillofac Surg. 2001;59: 445-7. https://doi.org/10.1053/joms.2001.21884 DOI: https://doi.org/10.1053/joms.2001.21884
Ulualp SO, Rodrigues SC, Hernandez J, Hay M. Bilateral atresia of the submandibular duct orifices. Am J Otolaryngol. 2007;28:184-6. https://doi.org/10.1016/j.amjoto.2006.06.020 DOI: https://doi.org/10.1016/j.amjoto.2006.06.020
Scher BL and Scher I. Case of imperforate submandibular ducts in an infant. Br Dent J. 1955;98:324-5.
Foretich EA, Cardo VA, Zambito RF. Bilateral congenital absence of the submandibular duct orifices. J Oral Surg. 1973;31:556-7.
Pownell PH, Brown OE, Pransky SM, Manning SC. Congenital abnormalities of the submandibular duct. Int J Pediatr Otorhinolaryngol. 1992;24:161-9. https://doi.org/10.1016/0165-5876(92)90142-C DOI: https://doi.org/10.1016/0165-5876(92)90142-C
Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D; CARE Group*. The CARE Guidelines: Consensus-based Clinical Case Reporting Guideline Development. Glob Adv Health Med. 2013;2(5):38-43. DOI: https://doi.org/10.7453/gahmj.2013.008
Rosow DE, Ward RF, April MM. Sialodochostomy as treatment for imperforate submandibular duct: a systematic literature review and report of two cases. Int J Pediatr Otorhinolaryngol. 2009;73:1613-5. https://doi.org/10.1016/j.ijporl.2009.03.007 DOI: https://doi.org/10.1016/j.ijporl.2009.03.007
Addante RR. Congenital cystic dilatation of the submandibular duct. Oral Surg Oral Med Oral Pathol. 1984;58:656-8. https://doi.org/10.1016/0030-4220(84)90029-X DOI: https://doi.org/10.1016/0030-4220(84)90029-X
Steelman R, Weisse M, Ramadan H. Congenital ranula. Clin Pediatr. 1998;37: 205. https://doi.org/10.1177%2F000992289803700309 DOI: https://doi.org/10.1177/000992289803700309
Redpath TH. Congenital ranula. Oral Surg Oral Med Oral Pathol. 1969;28:542-4. https://doi.org/10.1016/0030-4220(69)90262-x DOI: https://doi.org/10.1016/0030-4220(69)90262-X
Hoggins GS, Hutton JB. Congenital sublingual cystic swellings due to imperforate salivary ducts: Two case reports. Oral Surg Oral Med Oral Pathol. 1974;37:370-3. https://doi.org/10.1016/0030-4220(74)90109-1 DOI: https://doi.org/10.1016/0030-4220(74)90109-1
Kawahara K, Hotta F, Miyachi H, Shirozu T. Congenital dilation of the submandibular duct: report of a case. J Oral Maxillofac Surg. 2000;58:1170-2. https://doi.org/10.1053/joms.2000.9583 DOI: https://doi.org/10.1053/joms.2000.9583
Wada T, Yamamoto H, Ootsuki R, et al. Congenital ranula. Asian J Oral Maxillofac Surg. 2004, 16: 63-66. https://doi.org/10.1016/S0915-6992(04)80012-8 DOI: https://doi.org/10.1016/S0915-6992(04)80012-8
Capaccio P, Gaini LM, Pagani D, et al. Videosialoendoscopic assessment of bilateral atresia of the Wharton's duct orifice in an infant. J Pediatr Surg. 2007; 42: e5-e7. https://doi.org/10.1016/j.jpedsurg.2007.06.006 DOI: https://doi.org/10.1016/j.jpedsurg.2007.06.006
Pal K, Abdulla AM. Congenital imperforate submandibular duct in a newborn. Indian J Pediatr. 2007;74:687-8. https://doi.org/10.1007/s12098-007-0121-z DOI: https://doi.org/10.1007/s12098-007-0121-z
Basaran C, Ulu EMK, Donmez FY, et al. Unilateral congenital cystic dilatation of the submandibular gland duct: MDCT findings. Int J Pediatr Otorhinolaryngol. 2009;4:147-9. https://doi.org/10.1016/j.pedex.2008.10.004 DOI: https://doi.org/10.1016/j.pedex.2008.10.004
Ellegârd E, Kjellmer I. Atresia of the submandibular duct orifices: an unusual cause of feeding problems and failure to thrive in an infant. Acta Paediatr. 2010;99:1100-1. https://doi.org/10.1111/j.1651-2227.2010.01833.x DOI: https://doi.org/10.1111/j.1651-2227.2010.01833.x
Aronovich S, Edwards SP. A case of imperforate Wharton duct. J Oral Maxillofac Surg. 2014;72:744-7. http://dx.doi.org/10.1016/j.joms.2013.10.012 DOI: https://doi.org/10.1016/j.joms.2013.10.012
Hseu A, Anne P, Anne S. Congenital Atresia of Wharton's Duct. J Clin Diagn Res. 2016;10(2):MD01‐MD2.http://dx.doi.org/10.7860/JCDR/2016/17717.7279 DOI: https://doi.org/10.7860/JCDR/2016/17717.7279
Tanaka H, Takeuchi H, Shiga M, Hashimoto K. A case of atresia of the submandibular duct resulting in cystic swelling of the oral floor resembling a ranula. J Jap Soc Oral Surg. 2016;62:232-7. https://doi.org/10.5794/jjoms.62.232 DOI: https://doi.org/10.5794/jjoms.62.232
Pontes FSC, Souza LL, Pedrinha VF, Pontes HAR. Congenital Ranula: A Case Report and Literature Review. J Clin Pediatr Dent. 2018;42:454-7. https://doi.org/10.17796/1053-4625-42.6.9 DOI: https://doi.org/10.17796/1053-4625-42.6.9
Prosdócimo ML, Nogueira APB, Cavalcante MAA, et al. Congenital dilatation of the submandibular duct. Int J Pediatr Otorhinolaryngol. 2018;113:16-8. https://doi.org/10.1016/j.ijporl.2018.07.008 DOI: https://doi.org/10.1016/j.ijporl.2018.07.008
Chung YS, Cho Y, Kim BH. Comparison of outcomes of treatment for ranula: a proportion meta-analysis. Br J Oral Maxillofac Surg. 2019;57(7):620‐6. https://doi.org/10.1016/j.bjoms.2019.06.005 DOI: https://doi.org/10.1016/j.bjoms.2019.06.005
- Larissa Silva, Timilly Mayra Martins da Cruz, Moisés Willian Aparecido Gonçalves , Rafaela Nogueira Moreira Gonçalves , Cássio Roberto Rocha dos Santos , Ana Terezinha Marques Mesquita , Oral Blue Nevus two case reports and literature review. , Revista Estomatología: Vol. 29 No. 2 (2021)
- Moisés Willian Aparecido Gonçalves, Marina Rocha Fonseca Souza, Larissa Doalla de Almeida e Silva, Cássio Roberto Rocha dos Santos, João Luiz de Miranda, Jorge Esquiche León, Ana Terezinha Marques Mesquita , Nodular fasciitis on the zygomatic region: immunohistochemical analysis and literature review , Revista Estomatología: Vol. 30 No. 1 (2022)
Accepted 2022-03-09
Published 2022-02-07
This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.
Los autores/as conservan los derechos de autor y ceden a la revista el derecho de la primera publicación, con el trabajo registrado con la licencia de atribución de Creative Commons, que permite a terceros utilizar lo publicado siempre que mencionen la autoría del trabajo y a la primera publicación en esta revista.