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| CASE REPORT |
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| Year : 2019 | Volume
: 10
| Issue : 1 | Page : 53-56 |
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Congenital midline labial sinus of the upper lip
Anka Sharma, Subhash P Kumbhare, Ashita Kalaskar
Department of Oral Medicine and Radiology, Government Dental College and Hospital, Nagpur, Maharashtra, India
| Date of Web Publication | 15-Mar-2019 |
Correspondence Address:
Anka Sharma
Department of Oral Medicine and Radiology, Government Dental College and Hospital, Nagpur, Maharashtra
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/srmjrds.srmjrds_6_19
Congenital midline labial sinus is a rare condition affecting both the upper and lower lips. This anomaly has been reported in either gender but with a slightly increased predilection in females. It is known to occur either in isolation or in association with other congenital malformations such as cleft lip, cleft palate, van der Woude syndrome, median lip pit, brachidactilia, tongue tie, hypertelorism, notch of median vermillion border, nasal fistula, and abnormal hair pattern. In this article, we report a case of congenital midline sinus (without any associated congenital malformation) affecting the upper lip in a 12-year-old Indian male. Furthermore, a review of literature has been attempted on this condition. Keywords: Cellullitis, congenital midline labial sinus, upper lip
How to cite this article:
Sharma A, Kumbhare SP, Kalaskar A. Congenital midline labial sinus of the upper lip. SRM J Res Dent Sci 2019;10:53-6 |
| Introduction | |  |
Congenital midline labial sinus of the upper lip is a rare condition. This anomaly occurs either in isolation or in association with other congenital malformations such as cleft lip, cleft palate, and van der Woude syndrome.[1] The estimated prevalence of lower lip sinuses is about 0.00001% of the white population, but upper sinuses are even rarer.[2],[3] Till date, only one case has been reported in the Indian population.[4] We hereby report the second case in a 12-year-old Indian male with recurrent swelling of the upper lip since childhood.
| Case Report | | |
A 12-year-old male reported to the Department of Oral Medicine and Radiology with a chief complaint of recurrent episodes of pain and swelling of the upper lip since childhood. The patient would take over-the-counter drug, and the pain would subside while the swelling would reduce in size (but never resolved completely).
The present episode occurred 3 days back. The pain was sudden in onset and was followed by swelling of the upper lip. The patient had become febrile as well. He denied any history of trauma, and the medical as well as dental history was noncontributory. Family history was noncontributory. On further interrogation, the boy's grandmother revealed oozing of clear fluid from his lip when he was young.
On clinical examination, the patient was in acute pain (visual analog scale = 9) and was febrile (101°F).
A diffuse swelling was evident on the upper lip. No scar mark or draining sinus was evident. The local temperature was increased. It was tender on palpation and was soft firm in consistency. Lymphadenopathy was absent. Other extraoral findings were within normal limits.
On intraoral examination, the patient had a good oral hygiene. Two nodular swellings were evident on either side of the labial frenum. Suppuration point was evident on the swelling [Figure 1]a. It was tender and soft-firm on palpation. Anterior teeth had a normal morphology. The periodontium was healthy. | Figure 1: (a) The seemingly normal philtrum of the patient. (b) Nodular swelling on either side of frenum. Note the presence of suppuration points
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Due to acute pain, the patient refused any investigation and requested medications for prompt relief. He was prescribed a 5-day course of broad-spectrum antibiotics and analgesics with serratiopeptidase.
On the recall visit, the swelling and inflammation had reduced significantly. The nodular swellings had reduced in size. Vitality testing and intraoral periapical radiograph of the upper incisors revealed no abnormality, ruling out the possibility of a chronic dentoalveolar abscess.
A careful re-examination revealed a pinpoint depression over the philtrum [Figure 1]b. A 20 no. gutta-percha (GP) was inserted into it, and a radiograph was taken using the intraoral periapical (IOPA) film [Figure 2]a and [Figure 2]b. The radiograph did not reveal any foreign body. However, the presence of a sinus tract was confirmed. | Figure 2: (a) Insertion of a 20 no. gutta-percha point into the depression in the philtrum. (b) Radiograph depicting the gutta-percha point, suggesting the evidence of sinus tract
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A diagnosis of congenital midline sinus of the upper lip was made. The patient was recalled for further management by surgical excision of the sinus tract. However, the patient was lost to follow-up.
| Discussion | | |
Very few cases have been reported in the literature regarding this condition since the first ever case reported back in 1891 by Lannelongue and Menard.[5] In India, only one case has been reported so far, ours being the second.
Aoki et al. in 2011 proposed a classification for upper-lip sinuses.[6]
- Type 1: Midline sinus without accompanying anomalies
- Type 2: Midline sinus with accompanying anomalies
- Type 3: Lateral sinus with or without accompanying abnormalities.
The exact mechanism for this condition is not known. However, three theories have tried to establish its etiology:
- Invagination theory[7],[8],[9]
- Upper-lip sinuses are formed by the failure of ectodermal invagination of nasal placodes during the formation of frontonasal process
- Merging theory[10],[11]
- Sinuses are formed due to aberrations in the normal mesodermal merging process
- Fusion theory[12],[13],[14]
- Failure of complete fusion between the frontonasal and the maxillary process.
To the best of our knowledge, only 39 cases have been documented so far.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27],[28],[29],[30],[31],[32],[33],[34],[35],[36],[37],[38] Of these, 24 are from the Asian subcontinent, suggesting a predilection for this race. Only one case has been reported in the Indian population,[4] ours being the second. Furthermore, mostly affected are females, indicating a female predilection. In our case, the subject was a male contrary to the predilection. Majority of the cases have been reported in the 1–2nd decade, similar to our case. However, the minimum documented age at the time of presentation is 7 months,[20] while the maximum is 47 years.[34]
An array of associated congenital malformations can be seen in association with this anomaly. However, most of the cases are devoid of them as with our case. Usual complaint is recurrent swellings and discharge from the defect though some are asymptomatic. Sinus tracts are most commonly found. Fistulas are rarely encountered. Only one case reported the presence of both the sinus and fistula, where a cyst was also reported in association to the fistula.[12]
The mainstay of treatment for this condition is its early and prompt diagnosis followed by surgical excision of the sinus tract. Mere antibiotic coverage may simply suppress the infection and inflammation for some period of time, which may cause recurrence of symptoms at a later stage.
Patients with a history of recurrent cellulitis of either lip should be carefully examined for any evidence of a pit or a draining sinus and for the evidence of any congenital abnormality.
Sometimes, the pit is inconspicuous as was in our case. Hence, prior knowledge of this condition is a must.
A thorough case history followed by careful examination is usually sufficient to diagnose the condition. The confirmation of the sinus tract can be done using a GP point and an IOPA film. This method has many advantages:
- It is a quick and inexpensive technique
- GP points and IOPA films are readily available with any dental practitioner and hence can be performed even in a rural-based setup
- GP is an inert material. Hence, it is devoid of any allergic reaction (common with dyes).
| Conclusion | | |
Although congenital midline sinus of the upper lip is a rarest of the rare condition, its possibility should always be kept in mind by an oral diagnostician when diagnosing a patient with recurrent swellings of the upper lip. This article again emphasizes the role of thorough case history taking and careful examination in the art of diagnosis. One must always remember that “eyes can see what mind already knows.”
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The parents understand that name and initial will not be published and due efforts will be made to conceal patient identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]
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