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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 4  |  Issue : 4  |  Page : 173-176

Role of the pediatric dentist and prosthodontist in early cleft management: Presentation of two case reports


1 Department of Pedodontics and Preventive Dentistry, Thai Moogambigai Dental College and Hospital, Chennai, Tamil Nadu, India
2 Department of Prothodontics, SRM Dental College and Hospital, Kattankulathur, Chennai, Tamil Nadu, India

Date of Web Publication22-Jan-2014

Correspondence Address:
B N Rangeeth
987, TVS Colony, Anna Nagar West Extension, Chennai - 600 101
India
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DOI: 10.4103/0976-433X.125600

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  Abstract 

Cleft of lip and palate is one of the common congenital defects affecting the mankind. The management of this is based on a multidisciplinary approach with the parents who are often unaware of the condition until birth needing emotional support and professional guidance. The pediatric dentist is one of the constant entities of whose role starts from infancy through adolescence. This case report highlights the early role of the pediatric dentist in management of both syndromic and nonsyndromic cleft defects with an introduction on a few etiological factors and the genetics behind clefting.

Keywords: Cleft lip, cleft palate, feeding plate, nonsyndromic clefts, oral defects, Pierre robin syndrome


How to cite this article:
Rangeeth B N, Ahmed S, Cholan R, Russia M, Mohammed Raffi A J. Role of the pediatric dentist and prosthodontist in early cleft management: Presentation of two case reports. SRM J Res Dent Sci 2013;4:173-6

How to cite this URL:
Rangeeth B N, Ahmed S, Cholan R, Russia M, Mohammed Raffi A J. Role of the pediatric dentist and prosthodontist in early cleft management: Presentation of two case reports. SRM J Res Dent Sci [serial online] 2013 [cited 2020 Nov 28];4:173-6. Available from: https://www.srmjrds.in/text.asp?2013/4/4/173/125600


  Introduction Top


Research shows, learning to live with a change in appearance of one's face as a result of injury or disease is a difficult task. It is additionally challenging for children with congenital craniofacial conditions and their parents to adjust. [1],[2] Orofacial cleft is among the most common facial abnormalities, and the birth of a child with any of these and the prospect of a schedule of treatments that will continue into late adolescence puts enormous stress on the entire family. [3]

Nonsyndromic cleft lip with or without cleft palate (NSCL/P) is the most common congenital anomaly, with various prevalence rates among races with highest prevalence rate in Asian and lowest in African populations. [4] The etiology of orofacial cleft is multifactorial being both genetic and environmental. A number of significant breakthroughs have occurred with respect to the genetics of these conditions, in particular, characterization of the underlying gene defects associated with several important clefting syndromes. [5] Out of the several genes discovered recently, three of them - namely the T-box transcription factor-22 (TBX22) gene, poliovirus receptor like-1 (PVRL1) gene, and interferon regulatory factor-6 (IRF6) gene are responsible for causing X-linked cleft palate (Syndromic Cleft), cleft lip/palate-ectodermal dysplasia syndrome, and Van der Woude's (VWS) and popliteal pterygium syndromes, respectively. These genes are also implied in nonsyndromic cleft lip and palate. [6] Identification of a mutation in IRF6 is associated with an increase in the risk of having a child with CL/P from 4% to 6%, the risk of transmission of an isolated cleft, to 50%, the risk of transmission of a dominant Mendelian disorder like VWS. Studies done in Europe, Belgium, and America on the association of isolated CL/P with the IRF6 locus, a clear association was observed. This suggests that IRF6 also contributes to the occurrence of sporadic, isolated CL/P, even if no mutation in the gene can be identified. [7]

Jaju et al.[8] reported that 92% of the programs included the pediatric dentist in the multidisciplinary cleft palate team with the role extending from preventive, restorative to infant orthopedics Jaju et al.[8] reported that 92% of the cleft palate treatment protocols included the pediatric dentist in the multidisciplinary cleft palate team. The inclusion of pediatric dental support in the cleft team to identify 'at risk' patients and facilitate the provision of care through hospital, community, and general dental practitioner-based services has been shown to be effective. The patient with a cleft should be managed as "normal" but specific needs are to be incorporated into the stages of dental development and this article sheds light in an early stage of early management, which we feel is a key to success in the management of cleft lip and/or palate. [9],[10]


  Case Report Top


The first patient is a 43-day-old infant weighing 2.8 kg [Figure 1], born out of a consanguineous marriage. The child had been diagnosed with nonsyndromic cleft lip and palate and was under medical monitoring for the same. The second patient is a 56-day-old infant weighing 2.6 kg diagnosed with Pierre Robin Sequence [Figure 2] and [Figure 3] (syndromic cleft), was born out of nonconsanguineous marriage. For both the infants initially a naso-gastric tube had been used to aid in feeding, later it was removed and the parents were advised to dispense the breast milk into a feeding bottle and express the milk into the oral cavity. The patients were referred to the department for management and the parents expressed their concern on outflow of milk through the nose. Both were declared unfit for surgical management considering their birth weights. Our management strategy aimed to improve the physical fitness by increasing the feeding efficiency.
Figure 1: Intraoral view of a neonate diagnosed with nonsyndromic orofacial cleft

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Figure 2: Neonate diagnosed with Pierre Robin sequence referred to the Department of Pedodontics

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Figure 3: Special tray prepared for making an accurate impression

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The first step involved examination of the oral cavity followed by preliminary impression with a wax-based impression material and the model obtained is used for constructing an acrylic special tray [Figure 4]. This special tray roughly reproduces the features of the alveolus and the defect, which ensures a more accurate impression with a silicone-based impression material [Figure 5]. The model thus obtained from this impression is used to prepare a feeding plate [Figure 6] that will form a barrier preventing oroantral communication. Two holes are prepared to tie a length of floss that goes round the ears to stabilize the feeding plate. The infants were made to wear the feeding plate and the mother was asked to breast-feed the child [Figure 7] and [Figure 8]. The mothers were instructed about the method of usage, function, cleaning, and maintenance of feeding plate. Both infants exhibited excellent response after an initial setting in period of 60 minutes. An initial review after 2 days was done followed by a second review after 1 week and follow ups were suggested monthly. The infants showed excellent response that was shown by an increase in the weight making them eligible candidates for surgical intervention.
Figure 4: Accurate impression made with a silicone impression material

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Figure 5: Feeding plate made with heat polymerizing acrylic resin

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Figure 6: Neonate diagnosed with nonsyndromic orofacial cleft wearing the feeding plate

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Figure 7: Neonate diagnosed with Pierre Robin sequence wearing the feeding plate

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Figure 8: Intraoral view of the neonate diagnosed with Pierre Robin sequence

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  Discussion Top


Most parents are traumatized when a child is born with orofacial cleft and there are increased financial, social, and personal impacts before primary treatment is completed. The problems in coping is more in families with children having cleft lip and palate when compared to families with isolated cleft palate. [11] The quality of life is increasingly recognized as an important health outcome in people with surgically treatable conditions and satisfaction depended not only on surgical results but importantly on communication, empathy, expectations, postsurgical care, and discharge management. [12] The present trend of management involves reparative surgery within the 12 months of life. At this age the body weight varies between 5 and 10 kg and the whole blood volume between 400 and 700 ml. There was a direct relationship between decreased body weight and complications in surgery. [13]

Neonates with a cleft palate have difficulty in eating, which may lead to failure to thrive. [14] The oronasal communication diminishes the ability to create negative pressure, which is necessary for suckling. [15] To compensate, the baby presses the nipple between the tongue and the hard palate to squeeze out the liquids and milk, but this mechanism is insufficient if cleft is wide and the nipple gets trapped inside the defect. [16] The feeding process is also complicated by nasal regurgitation of food, excessive air intake that requires frequent burping and choking. [14],[17] The use of feeding tube which is started early in life includes soft tissue perforation. The feeding tubes are said to be responsible for most of the recorded perforations as the tissues are soft at first and the tissues become hard and stiff after several hours of use. [18] The feeding tube also produces rare complications such as urinary bladder perforation, pericardial sac perforation, and Enterobacteriaceae colonization [19],[20],[21] have been reported. Considering the complications that can be encountered, a safer and less invasive method is advisable for long-term management in relation to feeding the child with orofacial cleft. An evaluation of the feeding practices showed unsuccessful direct breast feeding and the use of acrylic plate was considered helpful by a majority of the study group. [22] Examination of the impact of age and lexical status at the time of primary palatal surgery on speech outcome of preschoolers with cleft palate suggested that children who were less lexically advanced and younger (mean age = 11 months) at the time of palatal surgery exhibited better articulation and resonance outcomes at 3 years of age. [23] Early management becomes a necessity as nonsyndromic clefts do affect the quality of life in school-age children and several limitations mostly affecting social role are seen. [24] Therefore there is a need for the early intervention by conservative means to decrease complications by increasing body weight and decreasing risk of complications in surgery.


  Conclusion Top


The pediatric dentist performs a dual role in both improving the personal impact as well as improving the surgical outcome. One of the reasons for personal impact, that is, effect of the anomaly in the orofacial complex, is the absence of proper feeding practices as we had seen with the parents of both the infants. The surgical outcome as mentioned earlier has a direct relationship with the body weight and this is also addressed by the use of the feeding plate. Considering risk of complications by use of a feeding tube the feeding plate is safe and hygienic. The pediatric dentist also facilitates the integration of oral hygiene and dental preventive regimens into the treatment protocol for these children in order to establish desirable habits and oral health prior to the provision of advanced reparative surgery and complex dental treatment.

 
  References Top

1.Rankin M, Borah GL. Perceived functional impact of abnormal facial appearance. Plast Reconstr Surg 2003;111:2140-6.  Back to cited text no. 1
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3.Chapados C. When a child is born with a cleft lip and palate. Can Nurse 1997;93:31-6.  Back to cited text no. 3
    
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7.Ghassibe M, Bayet B, Revencu N, Desmyter L, Verellen-Dumoulin C, Gillerot Y, et al. Orofacial clefting: Update on the role of genetics. B-ENT 2006;2 Suppl 4:20-4.  Back to cited text no. 7
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9.Udin RD. The pediatric dentist and the craniofacial anomalies team. Ear Nose Throat J 1986;65:305-10.  Back to cited text no. 9
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11.Kramer FJ, Baethge C, Sinikovic B, Schliephake H. An analysis of quality of life in 130 families having small children with cleft lip/palate using the impact on family scale. Int J Oral Maxillofac Surg 2007;36:1146-52.  Back to cited text no. 11
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12.Stone MB, Botto LD, Feldkamp ML, Smith KR, Roling L, Yamashiro D, et al. Improving quality of life of children with oral clefts: Perspectives of parents. J Craniofac Surg 2010;21:1358-64.  Back to cited text no. 12
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14.Goldberg WB, Ferguson FS, Miles RJ. Successful use of a feeding obturator for an infant with a cleft palate. Spec Care Dentist 1988;8:86-9.  Back to cited text no. 14
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15.Choi BH, Kleinheinz J, Joos U, Komposch G. Sucking efficiency of early orthopaedic plate and teats in infants with cleft lip and palate. Int J Oral Maxillofac Surg 1991;20:167-9.  Back to cited text no. 15
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16.Shprintzen RJ. The implications of the diagnosis of Robin sequence. Cleft Palate Craniofac J 1992;29:205-9.  Back to cited text no. 16
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17.Jones JE, Henderson L, Avery DR. Use of a feeding obturator for infants with severe cleft lip and palate. Spec Care Dentist 1982;2:116-20.  Back to cited text no. 17
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18.Boros SJ, Reynolds JW. Duodenal perforation: A complication of neonatal nasojejunal feeding. J Pediatr 1974;85:107-8.  Back to cited text no. 18
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19.Hanafy Eel D, Ashebu SD, Naqeeb NA, Nanda HB. Pericardial sac perforation: A rare complication of neonatal nasogastric tube feeding. Pediatr Radiol 2006;36:1096-8.  Back to cited text no. 19
    
20.Hurrell E, Kucerova E, Loughlin M, Caubilla-Barron J, Hilton A, Armstrong R, et al. Neonatal enteral feeding tubes as loci for colonisation by members of the Enterobacteriaceae. BMC Infect Dis 2009;9:146.  Back to cited text no. 20
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21.Mattar MS, al-Alfy AA, Dahniya MH, al-Marzouk NF. Urinary bladder perforation: An unusual complication of neonatal nasogastric tube feeding. Pediatr Radiol 1997;27:858-9.  Back to cited text no. 21
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22.Trenouth MJ, Campbell AN. Questionnaire evaluation of feeding methods for cleft lip and palate neonates. Int J Paediatr Dent 1996;6:241-4.  Back to cited text no. 22
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23.Chapman KL, Hardin-Jones MA, Goldstein JA, Halter KA, Havlik RJ, Schulte J. Timing of palatal surgery and speech outcome. Cleft Palate Craniofac J 2008;45:297-308.  Back to cited text no. 23
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24.Kramer FJ, Gruber R, Fialka F, Sinikovic B, Hahn W, Schliephake H. Quality of life in school-age children with orofacial clefts and their families. J Craniofac Surg 2009;20:2061-6.  Back to cited text no. 24
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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