Introduction
Among the oro-facial defects, cleft lip and palate (CLP) is the most
common and occurs in 4th to 12thweek of intra uterine life. Classification proposed by Veau’s divides
cleft palate into 4 main categories: Group I: Defects of Soft Palate
only, Group II: Defects involving hard and soft palate, Group III:
Defects involving soft palate to the alveolus usually involving lip and
Group IV: Complete bilateral clefts.(1) A systematic review by Panamota
et al. showed that prevalence of cleft lip and palate varies from 0.57
-1.57 per thousand live births.(2) The aetiology of cleft lip and palate
is multi-factorial and is influenced by both environmental and genetic
factors. Environmental factors include cigarette smoking, alcohol
intake, nutritional status of the mother such as vitamin and folic acid
deficiencies, obesity, diabetes mellitus etc. Genetic factors are
associated with various genes and genetic loci that are known to cause
isolated clefts- IRF6, ch8q24, vax1 and PAX7.(3) Strong correlation is
also seen in consanguineous marriages and family history. When the
neonate is born with CLP, the primary concern is feeding. Because of the
oro-nasal communication, it is difficult for the child to create strong
negative intra-oral pressure hence, difficult to extract milk while
suckling. When the neonate is born with CLP, feeding is compromised due
oro-nasal communication that poses difficulty in achieving strong
negative intra-oral pressure while suckling. Moreover, leakage of fluid
from the nasal cavity, chocking and milk reflux leads to infection of
middle ear.(4) CLP patients have difficulty in pronunciation of sound
like like ‘s’, ‘ch’, ‘sh’ and ‘j’ are altered, more so in isolated cleft
palate defects.
According to the Glossary of Prosthodontic terms an obturator is “a
maxillofacial prosthesis used to close a congenital or acquired tissue
opening primary of the hard palate and/ or contiguous alveolar or soft
tissue structure.” It forms a seal and blocks the communication between
the nasal and the oral cavity, helps in swallowing, prevents fluid
leakage and hyper nasality, and enhances speech. It blocks the
communication between the nasal and the oral cavity that assists in
speech, swallowing, prevention of fluid leakage and hyper nasality. To
reach the proposed parameter of weight and blood hemoglobin level in the
stipulated time, proper nutrition of the child is of utmost importance.
The present case report is an illustrative representation of
rehabilitation of 12-day old neonate where fabrication of feeding
obturator has been done using ethylene vinyl acetate sheet in a single
appointment.
Case report
A 12-days old neonate was referred to Department of Dentistry in a
tertiary care centre from the neonatal intensive care unit of the
institute.
On intra-oral examination, it was found that, the infant had cleft
palate (Veau classification class II) extending to the soft palate
(figure 1). Since the age of the child does not permit surgery in the
near future, a feeding obturator was decided as the treatment plan.
The family history did not reveal consanguineous marriage among the
parents. However, the family tree showed family members with cleft
(figure 2). Medical history of the mother showed pre-gestational
diabetes mellitus.
To avoid aspiration due to vomiting it was instructed not to feed the
child till 2 hours prior to the impression making. Since the size of the
palatal arch of the child was very small and was difficult to insert
even the smallest available stock tray, hence, the child’s stainless
steel feeding spoon (figure 3)was customized with acrylic extension for
making impression of the palate (figure 4). Impression of the palate was
made extending to the most posterior region of the cleft. While
impression making the child was awake, crying and without any
anaesthesia or any premedication. The head of the patient was tilted
downwards during impression making. A gauze piece of appropriate size,
attached to dental floss was stuffed along the borders of the defect
(figure 5). Impression material was loaded on the custom tray and was
hand moulded by the operator as the child could not perform proper
functional movements. The impression was made using fast setting rubber
base addition silicone impression material (Elite HD+ Putty Soft Fast
Set, Zhermack, Italy) (figure 6). After setting of the impression
material, it was retrieved from the child’s mouth. The mouth was
inspected for any broken or residual impression material. The cast was
poured in dental stone type IV. Deep undercuts were blocked in the
master cast with wax.
A feeding appliance was fabricated using 2mm thick, low density ethylene
vinyl acetate sheet using vacuum former machine (Biostar VI, USA) on a
single visit (figure 7). Thermal moulding was done for 60 seconds. It
was ensured that the sheet was properly adapted onto the palate and
ridges and extension into the vestibule was adequate. It was retrieved
from the cast, trimmed and polished before inserting in the patients
mouth. The borders were smoothened properly to avoid impingement and
ulcerations on the oral mucosa. After insertion the extensions were
checked, marked and trimmed accordingly. A floss was attached to the
feeding appliance (figure 7) to prevent the accidental aspiration and
easy retrieval of appliance. Then, the obturator was placed in the mouth
and bottle feeding was done (figure 8). The child could be easily fed
with appliance placed in his mouth. There was no leakage of fluid from
the nose. There was no obstruction on breathing. The parents were
demonstrated on inserting and removing the feeding obturator. They were
advised to clean the obturator properly with normal water, soap and soft
brush. It is instructed to remove the obturator at night. Also, proper
cleaning of the child’s oral cavity with wet cloth wrapped around the
finger was advised. Follow up after 24 and 72 hours were done. No
ulcerations were seen in the child’s oral cavity. On the day of delivery
of the appliance and each follow up the parents were trained on
insertion and removal of the obturator. A follow up after every 3 months
was suggested for refabrication to accommodate the growth of the palate.
Discussion
CLP are complex disorders with varying etiology. Failure of fusion of
fronto-nasal and maxillary processes causes cleft lip and that of
palatal shelves of the maxillary process leads to cleft palate.(5) The
incidences of cleft lip and palate are- cleft lip alone- 15%, cleft lip
and palate- 45% and isolated cleft palate- 40%. The major etiology of
cleft is mainly related to genetic factors, maternal risk factors,
environmental and teratogenic factors. Genetic factors include single
gene mutations, genomic locations, chromosomal abnormalities or
polygenic genes, environmental factors like phenytoin, valproic or
retinoic acid or any chemical substances consumed at the time of
pregnancy that might be teratogenic; maternal risk factors such as
alcoholism, smoking, addiction to drugs like benzodiazepens, diabetes,
maternal age more than 40 years etc.(6) it has been found that
gene-environment interaction plays an important role in the onset of the
deformity.(5) As in the present report the mother had history of
diabetes mellitus. Preconceptionally diabetes mellitus is a known risk
factor for oro-facial malformations like cleft. Mothers with history of
Type 1 and Type 2 diabetes mellitus have 3-4 times higher chances of
cleft. (7,8) Various studies have shown that congenital malformations
are 3-4 times more in mothers with pre- or gestational diabetes
mellitus.(7,9) The family history of the patient revealed cleft. The
family history up to 5 generations were charted. The pattern of
inheritance showed X- linked dominance.
Cleft palate surgeries are done in the
9th-18th month of child’s age.(10)
To combat the various problems associated with cleft various
modifications in feeding devices have been done such as Haber’s nipples.
However, even with these modifications chances of regurgitations and
reflux is there. The feeding obturator helps in feeding, positioning the
tongue away from the cleft allowing movement of segment towards each
other. Palatoplasty and pharyngoplasty are done at 12 months and 6 years
of age for speech enhancement in later years.
During fabrication of the feeding appliance or obturator selection of
impression tray, impression material and the patient positioning during
impression are important. Stock tray or custom trays are mostly used for
making impression. The use of stock tray requires two stages of
impression making- primary and final impression. This would require
multiple visits by the patient. In the present study, the appliance was
delivered in a single appointment, hence modified impression tray using
the patient’s feeding spoon was used. Acrylic extensions were made to
match the size of the child’s palate. The edges of the extension were
smoothened to avoid any irritation or abrasion to the mucosa. It helps
in easier and faster, single stage impression making customized
according to the size of the patient’s palate. Various impression
materials that can be used are irreversible hydrocolloids and
rubber-based impression materials. Poly-vinyl siloxane soft putty
viscosity impression material helps in making impression faster,
provides better handling and control over the flow of impression
material compared to irreversible hydrocolloid impression material.
Polyvinyl siloxane have more strength and elasticity hence, can be
retrieved from the defect without breakage of the impression material
into the defect. It provides good surface detail and allows retrieval of
multiple casts. While making the impression a gauze piece was placed in
the undercut of the defect to avoid the impression material from
engaging into deep undercuts causing problem in retrieval. While making
impression the child’s face was turned downwards to avoid asphyxiation
and the child was crying during the procedure ensuring a patent airway.
During impression making the child’s face was turned downwards to avoid
asphyxiation due to airways obstruction by the impression material. The
child was crying during impression making which ensures a patent airway.
Various materials that can be used for fabrication of obturator are auto
polymerizing self-cure acrylic resin, heat cure acrylic resin and vacuum
adapted ethylene vinyl acetate sheet. Self-cure and heat cure acrylic
resin are hard and not flexible. They provide a seal but may be
uncomfortable for the child due to its hard texture and there are more
chances of ulceration. A vacuum adapted low density ethylene vinyl
acetate sheet has been used for the fabrication of obturator. It is
light weight, moldable and offers a good fit over the palate. Its
adaptability is good, is soft, prevents ulcerations in the oral cavity
and requires less adjustments. Hence, the acceptance of the ethylene
vinyl acetate sheet obturator by the child is better. Keeping in view
Conclusion
The feeding of neonate is important for proper nutrition and weight
gain. The feeding appliance obturator provides benefits not only in
proper intake of food but also improves speech, prevents regurgitation
and infection of the middle ear. The modification of the feeding spoon
to a customized impression tray helps to obtain an accurate impression
in a short appointment. Ethylene vinyl acetate sheet provides benefits
of soft texture, better acceptability as well as fabrication in a single
appointment.
REFERENCES
1. Veau V. Paris: Masson & Cie; 1931. Division Palatine.
2. Panamonta V, Pradubwong S, Panamonta M, Chowchuen B. Global Birth
Prevalence of Orofacial Clefts: A Systematic Review. J Med Assoc Thai
2015;98:11-21.
3. Rahimov F, Jugessur A, Murray JC. Genetics of nonsyndromic orofacial
clefts. Cleft Palate Craniofac J 2012;49:73–91.
4. Freitas JA de S, Garib DG, Oliveira M, Lauris R de CMC, Almeida ALPF
de, Neves LT, et al. Rehabilitative treatment of cleft lip and palate:
experience of the Hospital for Rehabilitation of Craniofacial Anomalies
- USP (HRAC-USP) - Part 2: Pediatric Dentistry and Orthodontics. J Appl
Oral Sci 2012;20:268–81.
5. Candotto V, Oberti L, Gabrione F, Greco G, Rossi D, Romano M, et al.
Current concepts on cleft lip and palate etiology. J Biol Regul Homeost
Agents 2019;33:145-151.
6. Freitas Ja De S, Neves Lt Das, De Almeida Alpf, Garib Dg,
Trindade-Suedam Ik, Yaedú Ryf, Et Al. Rehabilitative treatment of cleft
lip and palate: experience of the Hospital for Rehabilitation of
Craniofacial Anomalies/USP (HRAC/USP) - Part 1: overall aspects. J Appl
Oral Sci 2012;20:9–15.
7. Kozma A, Radoi V, Ursu R, Bohaltea CL, Lazarescu H, Carniciu S.
Gestational diabetes mellitus and the development of cleft lip / palate
in newborns. Acta Endocrinol (Buchar) 2019;15:118–22.
8. Xu W, Yi L, Deng C, Zhao Z, Ran L, Ren Z, et al. Maternal
periconceptional folic acid supplementation reduced risks of
non-syndromic oral clefts in offspring. Sci Rep 2021;11:12316.
9. Kutbi H, Wehby GL, Moreno Uribe LM, Romitti PA, Carmichael S, Shaw
GM, et al. Maternal underweight and obesity and risk of orofacial clefts
in a large international consortium of population-based studies. Int J
Epidemiol 2017;46:190–9.
10.Vyas T, Gupta P, Kumar S, Gupta R, Gupta T, Singh HP. Cleft of lip
and palate: A review. J Family Med Prim Care 2020;9:2621-25.
FIGURE LEGENDS
Figure 1: Intra- oral view of cleft palate
Figure 2: Family tree of the infant
Figure 3: Feeding spoon
Figure 4: Modification in feeding spoon
Figure 5: Gauze piece with floss attached
Figure 6: Impression of patient
Figure 7: Obturator with floss attached
Figure 8: Bottle feeding with obturator