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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 2  |  Page : 133-135

Cleft palate with lateral oral synechiae presenting in late childhood: An extreme rarity


1 Department of Plastic Surgery, Medical College, Kolkata, West Bengal, India
2 Department of Plastic Surgery, The Institute of Post-Graduate Medical Education and Research, Kolkata, West Bengal, India

Date of Web Publication17-Aug-2015

Correspondence Address:
Dr. Prabir Kumar Jash
Department of Plastic Surgery, Green Building, 2nd Floor, Medical College, 88, College Street, Kolkata - 700 073, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2348-2125.162971

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  Abstract 

Congenital lateral oral synechia is a very rare anomaly which may be present in association with cleft palate and other facial abnormalities. Infants with lateral oral synechiae usually present very early in the neonatal period with restricted mouth opening and the resultant feeding difficulty, warranting early operative intervention. In the present article the authors report a 12-year-old child with lateral oral synechiae and cleft palate, presenting in late childhood and managed successfully by single-stage surgical correction.

Keywords: Cleft palate, congenital, lateral oral synechia


How to cite this article:
Jash PK, Chattopadhyay D, Rampuri V, Ahmed F. Cleft palate with lateral oral synechiae presenting in late childhood: An extreme rarity. J Cleft Lip Palate Craniofac Anomal 2015;2:133-5

How to cite this URL:
Jash PK, Chattopadhyay D, Rampuri V, Ahmed F. Cleft palate with lateral oral synechiae presenting in late childhood: An extreme rarity. J Cleft Lip Palate Craniofac Anomal [serial online] 2015 [cited 2019 Jun 19];2:133-5. Available from: http://www.jclpca.org/text.asp?2015/2/2/133/162971


  Introduction Top


The association of congenital oral synechia and cleft palate is an extremely rare syndrome. Lateral oral synechiae are defined as membranous adhesions between the floor of the mouth and the free margin of the cleft palate. Neonates with lateral oral synechiae typically present very early after birth because of restricted mouth opening and feeding difficulty, requiring early surgical intervention. In the present article the authors describe a case of lateral oral synechiae and cleft palate presenting in late childhood and managed successfully by single-stage surgical correction.


  Case Report Top


A 12-year-old boy was brought to the Out Patient Department for cleft palate. He was the first child of a healthy couple, born of nonconsanguineous marriage, and the antenatal period was uneventful. On examination he had a wide, complete bilateral cleft palate along with membranous bands connecting the margin of the palatal cleft with the floor of the mouth, lateral to the tongue bilaterally [Figure 1] and [Figure 2], which had been present since birth. These lateral oral synechiae were membranous, pink in color. They had broad attachments to the floor of the mouth measuring approximately 1.5 cm in width near the lingual side of first lower premolar tooth, and at the palatal margins (around hard palate-soft palate junction) the attachments narrowed to about 0.5 cm. The patient had no restriction of mouth opening. Examination of the ears, nose, extremities and genitalia did not reveal any anomaly. The developmental milestones were normal, and there was no evidence of mental retardation. Echocardiography and abdominal ultrasonography revealed normal visceral anatomy.
Figure 1: Cleft palate with lateral synechiae, frontal intraoral view


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Figure 2: Cleft palate with lateral synechiae, oblique intraoral view


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  Surgical Correction Top


Under general anesthesia, the membranous bands were excised. The band could be utilized minimally in nasal layer closure. However, with a narrow base of only 0.5 cm at palatal margins, the entire length of membranous band was not used. Only a centimeter of the membranous band was used for the same. The hard palate was closed by Bardach's two flap palatoplasty and the soft palate was repaired by intra-velar veloplasty. The postoperative result was satisfactory [Figure 3], and there were no complications. The child is doing well on follow-up after 3 months.
Figure 3: Postoperative picture after 3 weeks


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  Discussion Top


Hayward and Avery (1957) reported the first documented case of a lateral synechia between the floor of mouth and free margin of cleft palate. [1] Out of around 60 documented cases of oral synechiae till date, 52 were of lateral and other were of median variety. [2]

Oral synechia have been classified into five types:

  1. Alveolar synechia (synechia by cord-like adhesion of the alveolar mucosa on one or both sides of the upper and lower jaw);
  2. lateral synechia (synechia by a membranous adhesion on the hard palate and floor of the mouth, excluding the rear of the tongue);
  3. synechia in which the hard palate and tongue are partially involved;
  4. synechia in which the soft palate and tongue are widely involved, such that continuity is interrupted between the oral cavity and the pharynx; and
  5. synechia by a membranous adhesion between the hard palate and lower lip. [3]


Some of the reported cases of cleft palate lateral synechia (CPLS) syndrome were also accompanied by other facial anomalies like lower jaw hypoplasia, and shortened upper lip. [4] Significant restriction of mouth opening has been reported in majority of the cases of oral synechia. [2]

The etiology of oral synechiae is unclear and several theories have been proposed. According to Longacre, oral synechia results from the persistence of the buccopharyngeal membrane. [5] Kruger suggested that the mechanical effect of the tongue may contribute to the synechia between cleft palate margin and floor of the mouth. [6] Mathis postulated that when adhesion of the palatal shelf occurs during developmental stages, adhering epithelial rudiments lead to oral synechia. [7]

Patients with oral synechiae usually present very early in the neonatal period with difficulty in feeding due to restricted mouth opening. The treatment of CPLS syndrome is excision of the synechia and closure of the palatal cleft. [2] In present case, palatoplasty and synechia excision were performed in one stage. Donepudi et al. have demonstrated that the lateral synechia can provide additional tissue for surgical closure with less tension on the palatal flaps. [8] Following the same concept, the synechiae were utilized for palatal closure by the authors in the present case.


  Conclusion Top


In this article the authors describe a case of cleft palate with lateral oral synechiae, an extremely rare congenital anomaly. The present case also emphasizes the fact that congenital oral synechiae can present later in life if they are not severe enough to restrict normal mouth opening.

 
  References Top

1.
Hayward JR, Avery JK. A variation in cleft palate. J Oral Surg (Chic) 1957;15:320-4.  Back to cited text no. 1
[PUBMED]    
2.
Sybil D, Sagtani A. Cleft palate lateral synechia syndrome. Natl J Maxillofac Surg 2013;4:87-9.  Back to cited text no. 2
[PUBMED]  Medknow Journal  
3.
Ogino A, Onish K, Maruyama Y. Congenital oral synechia associated with cleft palate: Cleft palate medial synechia syndrome? Eur J Plast Surg 2005;27:338-40.  Back to cited text no. 3
    
4.
Fuhrmann W, Koch F, Schweckendiek W. Autosomal dominant inheritance of cleft palate and synechias between the palate and floor of the mouth or tongue. Humangenetik 1972;14:196-203.  Back to cited text no. 4
[PUBMED]    
5.
Longacre JJ. Congenital atresia of the oropharynx. Plast Reconstr Surg 1951;8:341-8.  Back to cited text no. 5
    
6.
Kruger GO. Textbook of Oral Surgery. 3 rd ed. St. Louis: Mosby; 1968. p. 537.  Back to cited text no. 6
    
7.
Mathis H. Case report of patient with difficulty in eating due to congenital syngnathia. German Dent J 1962;16:1167-71.  Back to cited text no. 7
    
8.
Donepudi SK, Stocks RM, Pivnick EK, Mineck C, Thompson JW. Cleft palate lateral synechia syndrome: An opportunity for unique surgical closure. Int J Pediatr Otorhinolaryngol 2009;73:861-6.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


This article has been cited by
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Japanese Journal of Oral and Maxillofacial Surgery. 2016; 62(10): 521
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  In this article
Abstract
Introduction
Case Report
Surgical Correction
Discussion
Conclusion
References
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