Advertisement

Midline Congenital Upper Lip Sinus: A Rare Case

  • Falak Chowdhary
  • Aniece Chowdhary
  • Zoya Chowdhary
  • Aamir Chowdhary
Clinical Report
  • 5 Downloads

Abstract

To present a rare case of a midline congenital upper lip sinus and its management. Background: A lip sinus is a rare condition, where a failure of the complete union during early stages of development leads to its formation. The prevalence of lower lip sinus is estimated to be about 0.00001% of the white population and that of upper lip sinus is even rarer. A congenital lip sinus maybe associated with syndromes such as; Von der Woude syndrome, or maybe present in isolation. A 25-year-old male presented to our clinic with intermittent swelling in the upper lip region. Clinical examination revealed a congenital midline lip sinus in the upper lip region, which had no intra-oral communication, and was managed by surgical excision. Till date, there has been a hand full of case reports of upper lip sinuses and fistulae, out of which only few are reported in India. We herein report a case of congenital upper lip sinus presenting as an intermittent swelling of the upper lip with its successful surgical management.

Keywords

Congenital Lip sinus Midline Upper lip pit 

Notes

Compliance with Ethical Standards

Conflict of interest

The authors declare that they have no conflict of interest.

References

  1. 1.
    Rajendran A, Sundaram S (2014) Shafer’s textbook of oral pathology, 7th edn. Elsevier Health Sciences APAC, Chatswood, pp 16–17. ISBN 978-81-312-3800-4Google Scholar
  2. 2.
    McKusick VA (1986) Commissural lip pits. Online Mendelian Inheritance in Man (OMIM). https://www.omim.org/entry/120500. Acessed 30 April 2018
  3. 3.
    Bakshi SS, Kiruba Shankar M, Gopalakrishnan S (2015) Midline upper lip sinus: a case report and review of literature. Oral Maxillofac Surg 19:217CrossRefPubMedGoogle Scholar
  4. 4.
    Rapini RP, Bolognia JL, Jorizzo JL (2007) Dermatology: 2—Volume set. Mosby, St. Louis. ISBN 1-4160-2999-0Google Scholar
  5. 5.
    Coccia CT, Bixler D (1967) Cleft lip, cleft palate, and congenital fistulas of the lower lip. Report of a familial occurrence. Oral Surg Oral Med Oral Pathol 24:246CrossRefPubMedGoogle Scholar
  6. 6.
    Cervenka J, Gorlin RJ, Anderson VE (1967) The syndrome of pits of the lower lip and cleft lip and/or palate. Genetic considerations. Am J Hum Genet 19:416PubMedPubMedCentralGoogle Scholar
  7. 7.
    Hili S, Wong KY, Goodacre T (2016) Isolated midline upper lip pit. BMJ Case Rep.  https://doi.org/10.1136/bcr-2016-215496 PubMedPubMedCentralCrossRefGoogle Scholar
  8. 8.
    James L, Veerabasvaiah BT, Ghouse N (2015) Congenital upper lip pit: a rare case report. J Indian Acad Oral Med Radiol 27:311CrossRefGoogle Scholar
  9. 9.
    Rifaat M, Jackson IT, Celebilor O (1998) The lateral upper lip sinus: case report and review of literature. Eur J Plast Surg 21:98CrossRefGoogle Scholar
  10. 10.
    Parcelier A, Lacoste A (1914) Fistule congenitale de la levre superieure. Arch Gen Chir Paris 8:292Google Scholar
  11. 11.
    Feurer G (1893) Angeborene oberlippenfistel. Arch Klin Chir 46:35Google Scholar
  12. 12.
    Miller CJ, Smith JM (1980) Midline sinus of the upper lip and a theory concerning etiology. Plast Reconstr Surg 65:674CrossRefPubMedGoogle Scholar
  13. 13.
    Eppley BL, Sadove AM, Goldenberg J (1992) Philtral fistula in median cleft lip: cause and effect or coincidence? Ann Plast Surg 29:263CrossRefPubMedGoogle Scholar
  14. 14.
    Al-Qattan MM (2000) Congenital midline sinus of the upper lip. Ann Plast Surg 44:76CrossRefPubMedGoogle Scholar
  15. 15.
    Grenman R, Salo H, Rintala A (1985) Midline sinuses of the upper lip. Case report. Scand J Plast Reconstr Surg 19:215CrossRefPubMedGoogle Scholar
  16. 16.
    Parisier SC, Birken EA (1973) Congenital midline sinus of the upper lip. Arch Otolaryngol 97:259CrossRefPubMedGoogle Scholar
  17. 17.
    Mackenzie KI (1970) Mid-line sinus of the upper lip. J Laryngol Otol 84:235CrossRefPubMedGoogle Scholar
  18. 18.
    Bartels RJ, Howard RC (1973) Congenital midline sinus of the upper lip. Case report. Plast Reconstr Surg 52:665CrossRefPubMedGoogle Scholar
  19. 19.
    Kriens O, Schmidt H, Mueller-Driver O (1973) Congenital lateral fistula of upper lip. A case report. J Maxillofac Surg 1:122CrossRefPubMedGoogle Scholar
  20. 20.
    Fok D, Kua EHJ, Por YC (2015) Congenital midline sinus of the upper lip. Singapore Med J 56(6):e107CrossRefPubMedPubMedCentralGoogle Scholar
  21. 21.
    Aoki M, Sakamoto Y, Nagasao T, Miyamoto J, Kishi K (2014) Classification of congenital midline upper lip sinuses: a case report and review of the literature. Cleft Palate Craniofac J 51:154CrossRefPubMedGoogle Scholar
  22. 22.
    Menendez OR (1969) Congenital pit of the upper lip. Oral Surg Oral Med Oral Pathol 27:441CrossRefGoogle Scholar
  23. 23.
    Holbrook LA (1970) Congenital midline sinus of upper lip. Br J Plast Surg 23:155–160CrossRefPubMedGoogle Scholar
  24. 24.
    Urade M, Tofani I, Igarashi T, Morioka S, Nishimura K, Usui M et al (1984) Congenital midline sinus of the upper lip: report of a case. J Osaka Univ Dent Sch 24:59PubMedGoogle Scholar
  25. 25.
    Raibagkar S, Vyas UH (1986) Lateral congenital sinus of the upper lip. Indian J Plast Surg 19:50–52Google Scholar
  26. 26.
    Katou F, Motegi K (1989) Congenital midline sinus of the upper lip. Report of a case. Int J Oral Maxillofac Surg 18:237CrossRefPubMedGoogle Scholar
  27. 27.
    Sakamoto H, Imai Y, Asakura A (1992) Congenital midline sinus of the upper lip. Report of a case. Int J Oral Maxillofac Surg 21:10CrossRefPubMedGoogle Scholar
  28. 28.
    Mizuki H, Shimizu M, Danjo T (1993) Congenital fistula with an island of vermilion-epithelium in the paramidline of the upper lip: report of a case. J Craniomaxillofac Surg 21:22CrossRefPubMedGoogle Scholar
  29. 29.
    Asahina I, Sakakibara T, Miyashin M, Tachikawa N, Enomoto S (1997) Congenital midline sinus of the upper lip: case report and review of literature. Cleft Palate Craniofac J 34:83CrossRefPubMedGoogle Scholar
  30. 30.
    Illing HM, Field D, McNamara CM, Sandy JR (1999) Congenital sinus of the upper lip. A case report. Int J Oral Maxillofac Surg 28:29CrossRefPubMedGoogle Scholar
  31. 31.
    Nagasao T, Urushidate S, Yokoi K, Yotsuyanagi T, Sawada Y (2000) A case of congenital midline sinus of the upper lip. Eur J Plast Surg 23:289CrossRefGoogle Scholar
  32. 32.
    Sumitomo S, Ogawa S, Hirai K, Takai Y (2002) Congenital sinus of the upper lip with idiopathic precocious puberty. Oral Dis 8:308CrossRefPubMedGoogle Scholar
  33. 33.
    Sancho MA, Albert A, Cusi V, Grande C, Aguilar C, Morales L (2002) Upper lip fistulas: three new cases. Cleft Palate Craniofac J 39:457CrossRefPubMedGoogle Scholar
  34. 34.
    Lee TJ, Bin CW, Koh CS (2003) Congenital sinus of upper lip. J Korean Cleft Palate Craniofac Assoc 4:45Google Scholar
  35. 35.
    Charrier JB, Rouillon I, Roger G, Denoyelle F, Collon S, Garabedian EN (2006) Congenital isolated midline sinus of the upper lip: clinical and embryological approaches. Cleft Palate Craniofac J 43:488CrossRefPubMedGoogle Scholar
  36. 36.
    Sen C, Agir H, Isken T, Alagoz S, Karadeniz E, Iscen D (2006) Congenital midline upper lip sinus. J Craniofac Surg 17:810CrossRefPubMedGoogle Scholar
  37. 37.
    Nakano Y, Somiya H, Shibui T, Uchiyama T, Takano N, Shibahara T, Hashimoto S (2010) A case of congenital midline fistula of the upper lip. Bull Tokyo Dent Coll 51:31CrossRefPubMedGoogle Scholar
  38. 38.
    Salati SA, Al Aithan B (2012) Congenital median upper lip fistula. APSP J Case Rep 3:11PubMedPubMedCentralGoogle Scholar
  39. 39.
    Bhatnagar Ankur, Musa Osman, Gildiyal JP, Pandey M (2012) Congenital lateral upper lip sinus: a rare case. Indian J Surg 74:344CrossRefPubMedPubMedCentralGoogle Scholar
  40. 40.
    Anicete RC, Khoo I, Tan HKK (2014) Congenital midline upper lip sinus: a case report. Int J Pediatr Otorhinolaryngol e9:18CrossRefGoogle Scholar
  41. 41.
    Rohart J, Nicot R, Myon L, Elbaz J, Raoul G, Ferri J (2015) Labial recurrent swelling revealing median congenital upper-lip fistula. Rev Stomatol Chir Maxillofac Chir Orale 116:315PubMedGoogle Scholar
  42. 42.
    Xu Y, Chen R, Mu Y, Zheng Z (2016) Congenital midline sinus of the upper lip. J Craniofac Surg 27:1306CrossRefPubMedGoogle Scholar
  43. 43.
    Alam M, Hasan SA, Hashmi SF, Singh PK (2017) Isolated congenital midline upper lip sinus in a 5-year old child: a rarity. Indian J Otolaryngol Head Neck Surg 69:568CrossRefPubMedGoogle Scholar

Copyright information

© Association of Otolaryngologists of India 2018

Authors and Affiliations

  1. 1.ENT ClinicJammuIndia
  2. 2.Indira Gandhi Government Dental College and HospitalJammuIndia

Personalised recommendations