Ranula, Its Etiopathogenesis And Management; A Systematic Review

Aspects of etiopathogenesis, origin, and management remain debatable. The rationale of our study was to eliminate the discrepancies pertaining to the etiopathogenesis and management of ranula. Our objective was to determine the etiopathogenesis of ranula by systematically reviewing the available literature and to establish the best treatment protocol for ranula (intraoral and plunging). Case series, case reports, and observational and longitudinal studies referring to the etiology, pathogenesis, and management of ranula were included, and the previously published review articles were excluded from our study. We performed an electronic search from the year of inception of the database to December 2019. PubMed, Science direct, and Google Scholar databases were explored without restrictions of language or publication date. The search algorithm was Ranula {Mesh Major topic} and {Etiopathogenesis}, Ranula {Mesh Major topic} and {Management}. A total of 64 full-text articles were assessed. We concluded that the presence of the Bartholin duct might be a possible cause of intraoral ranula development, indicating that it originates from greater sublingual gland. Intraoral and plunging ranula are traumatic extravasation pseudocysts. Congenital ranula are histopathologically classified retention cysts lined with stratified squamous epithelium. The mylohyoid muscle exhibits dehiscence, which is commonly present in the anterior two-third part from which a part of the sublingual gland can be herniated or mucus can extravasate, leading to the formation of plunging ranula. Transoral excision with complete excision of ipsilateral sublingual gland remains the gold standard treatment of intraoral ranula and gold standard surgical treatment for plunging ranula. For mixed ranula, the treatment includes transoral excision of the sublingual gland along with the drainage of cyst.

PDF

___

1. Mcmurrich J P. The mouth and its organs.Manual of human embryology ,F Keibel and F P Mall ,editors,Phiadelphia,Lippincott Co.1912;2:347-349.
2. Thoma K. A Contribution to the knowledge of the development of the Submaxillary and Sublingual salivary glands in human embryos. J Dent Res. 919; 1: 95-143. [Crossref]
3. Leppi TJ. Gross anatomical relationships between primate Submandibular and Sublingual salivary glands. J Dent Res. 1967; 46: 359-365. [Crossref]
4. McGurk M, Eyeson J, Thomas B, Harrison JD. Conservative treatment of oral ranula by excision with minimal excision of sublingual gland: Histologic support of traumatic etiology. J Oral Maxillofac Surg.2008;66(1):2050-2057 [Crossref]
5. Mun SJ, Choi HG, Kim H, et al. Ductal variation of the sublingual gland: A predisposing factor of ranula formation. Head and Neck. 2014; 36(4): 540-544. [Crossref]
6. Zhang B, Yang Z, Zhang RM, J, et al. Are the patients with anatomic variation predisposed to ranula formation? Int J Pediatr Otorhinolaryngol. 2016; 83: 69-73. [Crossref]
7. Rayne J. Plunging ranula report of case. Br J Oral Surg .1973; 11: 139-14.2 [Crossref]
8. de Visscher JG, van der wal KG, de Vogel PL. The plunging ranula pathogenesis ,diagnosis and management. J Craniomaxillofac Surg.1989; 17(4): 182-185. [Crossref]
9. Olasoji HO, Tahir AA, Arotiba GT. Plunging ranula :A report of two cases. EAMJ. 2002; 79: 51-53. [Crossref]
10. Pandit RT, Park AH. Management of paediatric ranula. Otolayngol Head Neck Surg. 2002; 127: 115-118. [Crossref]
11. Hidaka H, Oshima T, Kakehata S, et al. Two cases of plunging ranula managed by intraoral approach. Tohoku J Exp Med. 2003; 200: 59-65. [Crossref]
12. Ghani NA, Ahmad R, Rahman RA, Yunus MR, Putra SHA, Ramli R. A retrospective study of ranula in two centres in Malaysia. J Maxillofac Oral Surg. 2009; 8(4): 316-319.[Crossref]
13. Arunachalam P, Priyadarshani N. Recurrent plunging ranula. J Indian Assoac Padiatr Surg. 2010; 15(1): 36-38. [Crossref]
14. Samant S, Morton RP, Ahmad Z. Surgery for plunging ranula the lesson not yet learned?. Eur Arch Otorhinolaryngol. 2011; 268:1513-1518. [Crossref]
15. Suresh BV, Vora SK. Huge plunging ranula. J Maxillofac Oral Surg. 2012; 11(4): 487-490. [Crossref] 16. Mustafa AB, Bokhari K, Luqman M, Hameed SM, Kota Z. Plunging ranula: An interesting case report. OJST. 2013; 3: 118-121. [Crossref]
17. Kim SH, Huh KH, An CH, Park JW, Yi WJ. Giant plunging ranula a case report. Imaging Sci Dent. 2013; 43: 55-58. [Crossref]
18. McKinstry S, Lewis C. NZMJ. 2013; 126: 81-86 [Crossref]
19. Olojede ACO, Ogundana OM, Emeka CI, et al. Plunging ranula: Surgical management of case series and literature review. Clin Case Rep. 2017; 6: 109-114. [Crossref]
20. Kamlakaran A, Jayaraman B, Balasubramanian S, Thirunavukkarasu R, Ramakrishnan B. Plunging ranula in 78 year old male-a rare case report. J Clin Exp Dent. 2018; 10(1): 92-95. [Crossref]
ram I, Cankaya H, Casken H, Faruk A, Kiris M. Paediatric intraoral ranula.an analysis of nine cases. Tohoku J Exp Med. 2005; 205: 151-155. [Crossref]
22. Date A, Padhye M, Desai R, Hire A. Simultaneous bilateral ranula in an edentulous patient. Rare presentation with a brief review of literature. 2016;18(4): 133-136.
23. Harrison JD. Salivary mucoceles. Oral surg Oral PathOral Med Oral Pathol. 1975; 39: 268. [Crossref]
24. Harrison JD, Kim A, Ali SA, Mortan RP. Postmortem investigation of mylohyoid hiatus and hernia: Aetiological factors for plunging ranula. Clin Anat. 2013; 26: 693-699. [Crossref] 25. Redpath TH. Congenital ranula. Oral Surg,Oral Med,Oral Path,Oral Radiol.1969; 28(4): 542-544. [Crossref]
26. Hoggins GS, Hutton GB. Congenital sublingual cystic swelling due to imperforate salivary ducts. Oral Surg.1974; 37: 370. [Crossref]
27. Pownell PH, Brown OE, Pransky SM, Manning SC. Congenital abnormalities of submandibular duct. Int J Paediatr Otorhinilayngol.1992; 24(2): 161-169. [Crossref]
28. Moya JMF, Sulzberger SC, Recasens DJ, Ramos C, Sanz R, Tejerizo GP. Antenatal diagnosis and management of a ranula. Ultrasound Obstet Gynecol. 1998; 11: 147-148. [Crossref]
29. Ugboko VI,Hassan O,Prasad S,Amole AO. Congenital ranula a report of two cases. ORL. 2002; 64: 294-296. [Crossref]
30. Onderoglu L, Saygan B, Deren O et al. Preanatal diagnosis of ranula at 21 weeks of gestation. Ultrasound Obstet Gynecol. 2003; 22: 399-401. [Crossref]
31. Kolker MT, Batti JS, Schoem SR. The ex utero intrapartum treatment procedure for congenital ranula in a jehovah’s witness. Otolayngol Head Neck Surg. 2004; 130: 508-510. [Crossref]
32. Cavalcante ASR, Rosa LEB, Costa NC, Hatakeyama M, Anbinder AL. Congenital ranula; A case report. J Dent Child. 2009; 76(1): 78-81.
33. Marques MI, Morais S, Coutinho S, de Castro O, Rei AI. Ranula congenita. Acta Med Port. 2010; 23(15): 937-940.
34. Soni A, Suyal P, Suyal A. Congenital ranula in newborn; A rare presentation. Indian J Otolaryngol Head Neck Surg. 2012; 64(3): 295-297. [Crossref]
35. Singh GB, Rai AK, Arora R, Garg S, Abbey P, Shukla S. A rare case of congenital simple cystic ranula in a neonate. Case Rep Otolayngol. 2013. [Crossref]
36. Mneimneh S, Barazi R, Rajab M. A rare case of congenital ranula in an infant. Case Report Otolaryngol. 2016. [Crossref]
37. Pant N, Verma AK, Khan T, Wakhlu A. Ectopic salivary gland cyst in neck: Association with congenital plunging ranula. J Indian Assaoc Padiatr Surg. 2017; 22(4): 251-253. [Crossref]
38. Morestein H. Pathogenie de la grenouillete sus-hyoidienne. Gazette des hopitaux civils et militaries .1897; 22: 529-533.
39. Gaughran GRL. Mylohyoid boutonniere and sublingual bouton. J Anat .1963; 97(4): 565-568.
40. Engel JD, Harn SD, Cohen DM. Mylohyoid herniation: Gross and histologic evaluation with clinical correlation. Oral Surg Oral Med Oral Pathol .1987; 63: 55-59. [Crossref]
41. Jain P, Jain R, Mortan RP, Ahmad Z. Plunging ranula: High resolution ultrasound for diagnosis and surgical management. Eur Radiol. 2010; 20: 1442-1449. [Crossref]
42. Jain P, Jain R. Types of sublingual gland herniation observed during sonography of plunging ranulas. J Ultrasound Med. 2014; 33: 1491-1497. [Crossref]
43. Lee YJ, Lee HY, Kim HJ, Jeong HS, Kim YK, Cha J. Plunging ranula revisited a CT study with emphasis on a defect of mylohyoid muscle as the primary route of lesion propagation. KJR. 2016; 17(2): 264-270. [Crossref]
44. Schneider W, Fulgenzi A, Lupovitch A. Submental herniation of sublingual gland, report of case. J Oral Surg.1972; 30: 824-826. 45. Braun TW, Sotereanos GC. Cervical ranula due to an ectopic sublingual gland. J Maxillofac Surg .1982; 10: 260-277. [Crossref]
46. Takimoto T, Masuda K, Nakai Y. Cervical ranula arising from a projection of the sublingual gland through the mylohyoid muscle. J Oral Maxillofac Surg.1989; 47: 1197-1198. [Crossref]
47. Ichimura K, Ohta Y, Tayama N. Surgical management of the plunging ranula a review of seven cases. J Layngol Otol. 1996; 110: 554-556. [Crossref]
48. Mortan RP, Ahmad Z, Jain P. Plunging ranula: Congenital or acquired? Otolaryngol HeadNeck surg. 2010; 142: 104-107. [Crossref]
49. Kuryabaschi T, Ida M, Yasumoto M. MRI of ranulas. Neuroradiology. 2000; 42: 917-922. [Crossref]
50. Li J, Li J. Correct diagnosis for plunging ranula by magnetic resonance imaging. ADJ. 2014; 59: 264-267. [Crossref]
51. Steelman R, Weisse M, Ramadan H. Congenital Ranula. Clin Pediatr. 1998; 37(3): 205-206. [Crossref]
52. Bernhard MK, Huckel D, Hamala D. Congenital ranula in a newborn. [Article in German] HNO. 2007; 55(5): 388-91. [Crossref]
53. Zhi K, Wen Y, Ren W, Zhang Y. Management of infant ranula. Int J Pediatr Otorhinolaryngol. 2008; 72: 823-826. [Crossref]
54. Bonet C, Mahmud J, Matinez M, Penarrocha MA. Congenital oral mucoceles: Presentation of four new clinical cases. An Paediatr Barc. 2011; 75: 424-425. [Crossref]
55. Mortan RP, Bartley JR. Simple sublingual ranulas: Pathogenesis and management. J Otolaryngol. 1995; 24: 253-254.
56. DelhamAC, Cunha RF, Vieira AE, Riebeiro LL. Treatment of mucous retention phenomenon in children by micromarsupialization technique. Case reports Paediatric Dent. 2000; 22(2): 155-158.
57. Sandirini FA, Santana Filho M, Rados PV. Ranula management : Suggested modification in the micromarsupialization technique. J Oral Maxillofac Surg. 2007; 65(7): 1436-1438. [Crossref] 58. Raj PP, Zarina R, Deepa K. Micromarsupialization of sublingual ranula. IOSRJAMS. 2019; 18(4): 67-73.
59. Zhao YF, Jia J, Jia Y. Complications associated with surgical management of ranulas. J Oral Maxillofac Surg. 2005; 63(1): 51-54. [Crossref]
60. Watanabe K, Nakajima H, Tomiyana S, Yagi T, Jinnouchi K. Local injection of OK-432in the treatment of ranula: A case report. Ear Nose Throat J. 2002; 81(2): 97-98. [Crossref]
61. Lee HM, Lim HN, Kong HJ, et al. Treatment of ranula in paediatric patients with intraoral injection of OK432. Laryngoscope. 2006; 116(6): 966-969. [Crossref]
62. Yoshizama K, Moroni A, Kawashori S, Ueki K. A case of sublingual ranula that reported successfully to localized injection treatment with OK432 after healing from drug induced hypersensitivity syndrome. 2016. [Crossref]
63. Garofalo S, Briganti V,Cavallaro S,Pepe E,Prete M,Suteu et al.Nickel gluconate mercurious heel potentised swine organ preparation : A new therapeutic approach for the primary treatment of paediatric ranula and intraoral mucocele. IJPORL. 2007; 71(2): 247-255. [Crossref]
64. Shehata EA, Hassan HS. Surgical treatment of ranula. Comparison between marsupialization and sublingual sialoadenectomy in paediatric patients. AOPS. 2008; 4: 89-93.
65. Fukase S, Ohta N, Inamura K, Aoyagi M. Treatment of ranula with intracystic injection of the streptococcal preparation of OK432. Ann Otol Rhino Laryngol. 2003; 112(3): 214-220. [Crossref]
66. Rho MH, Kim DW, Kwon JS, et al. OK432 sclerotherapy of plunging ranula in 21 patients: It can be a substitute for surgery. AJNR. 2006; 27: 1090-1095.
67. Ryu K, Lee JH, Lee JY, et al. Ethanol ablation of ranulas: Short term follow up results and clinicoradiological factors for successful outcome. AJNR. 2017; 38(9): 1794-1798. [Crossref]
68. Huang SF, Liao CT, Chin SC, Chen IH. Transoral approach for plunging ranula-10 years experience. Laryngoscope. 2010(1); 120: 53-57. [Crossref]