View of Feeding and swallowing disorders in children with clefts in the orofacial region

(1)

1 University Rehabilitation Institute Republic of Slovenia, Ljubljana, Slovenia

2 Department of Maxillofacial and Oral Surgery, Division of Surgery, University Medical Centre Ljubljana, Ljubljana, Slovenia

3 Department of

Otorhinolaryngology and Cervicofacial Surgery, University Medical Centre Ljubljana, Ljubljana, Slovenia

4 Faculty of Medicine, University of Ljubljana, Ljubljana, Slovenia Correspondence/

Korespondenca:

Nika Jelenc, e: nika.

jelenc@ir-rs.si Key words:

orofacial clefts; feeding and swallowing problems;

questionnaire; risk factors;

interventions Ključne besede:

razcepi v orofacialnem področju; težave s hranjenjem in požiranjem;

vprašalnik; dejavniki tveganja; obravnava Received: 27. 10. 2018 Accepted: 24. 3. 2019

eng slo element

en article-lang

10.6016/ZdravVestn.2892 doi

27.10.2018 date-received

24.3.2019 date-accepted

Stomatology Stomatologija discipline

Original scientific article Izvirni znanstveni članek article-type

Feeding and swallowing disorders in children

with clefts in the orofacial region Motnje hranjenja in požiranja pri otrocih z razcepi v orofacialnem področju

article-title Feeding and swallowing disorders in children

with clefts in the orofacial region Motnje hranjenja in požiranja pri otrocih z razcepi v orofacialnem področju

alt-title orofacial clefts, feeding and swallowing prob-

lems, questionnaire, risk factors, interventions razcepi v orofacialnem področju, težave s hranjenjem in požiranjem, vprašalnik, dejavniki tveganja, obravnava

kwd-group

The authors declare that there are no conflicts

of interest present. Avtorji so izjavili, da ne obstajajo nobeni

konkurenčni interesi. conflict

year volume first month last month first page last page

2019 88 9 10 405 414

name surname aff email

Nika Jelenc 1 nika.jelenc@ir-rs.si

name surname aff

Andreja Eberlinc 2

Irena Hočevar Boltežar 3,4

eng slo aff-id

University Rehabilitation Institute Republic of Slovenia, Ljubljana, Slovenia

Univerzitetni rehabilitacijski inštitut Republike Slovenije – Soča, Ljubljana, Slovenija

1

Department of Maxillofacial and Oral Surgery, Division of Surgery, University Medical Centre Ljubljana, Ljubljana, Slovenia

Klinični oddelek za maksilofacialno in oralno kirurgijo, Kirurška klinika, Univerzitetni klinični center Ljubljana, Ljubljana, Slovenija

2

Department of

Otorhinolaryngology and Cervicofacial Surgery, University Medical Centre Ljubljana, Ljubljana, Slovenia

Klinika za otorinolaringologijo in cervikofacialno kirurgijo, Univerzitetni klinični center Ljubljana, Ljubljana, Slovenija

3

Faculty of Medicine, University Medicinska fakulteta, Univerza v 4

Feeding and swallowing disorders in

children with clefts in the orofacial region

Motnje hranjenja in požiranja pri otrocih z razcepi v orofacialnem področju

Nika Jelenc,¹ Andreja Eberlinc,² Irena Hočevar Boltežar^3,4

Abstract

Background: Orofacial clefts are the most common congenital craniofacial malformations. Chil- dren with orofacial clefts do not only have problems with speech, but also with feeding and swallowing. Alternative methods of feeding need to be introduced in severe cases. Until now there has not been conducted a single study regarding feeding and swallowing problems in children with orofacial clefts in Slovenia.

Methods: The aim of the study was to determine the prevalence and the type of feeding and swallowing problems in children with orofacial clefts, aged between five months and six years.

An anonymous questionnaire for parents was created, containing questions about the type of the child’s orofacial cleft, child’s possible feeding, swallowing and speech problems, and special feeding gadgets. The questionnaire was initially tested in a pilot study during routine medical follow-up visits. Afterwards, it was sent to 93 parents of children with clefs, born in a period of three consecutive years. A descriptive statistics was used to present the prevalence of feeding and swallowing problems in children with orofacial clefts. The factors correlated with feeding problems were analysed.

Results: Almost three-quarters of children had problems with feeding immediately after birth;

problems with feeding later decreased, but they were still present in one quarter of all cases until the completed surgical treatment. Most difficulties occurred due to nasal regurgitation, excessive air intake while bottle feeding, and prolonged feeding time. Children with cleft palate with or without cleft lip had significantly more feeding problems in comparison to children with cleft lip only. Most parents used special feeding bottles and were generally satisfied with their use. More than a half of the parents believed that they had received too little information about feeding before and immediately after the child’s birth.

Conclusion: Despite the well-organized multidisciplinary care of children with orofacial clefts in Slovenia, parents still miss the professional counselling about feeding their children. Therefore, an additional education of nurses working in maternity hospitals will be necessary in the future.

Considering the high prices of special feeding gadgets for children with orofacial clefts, covering of their costs by the health insurance would be welcome.

Izvleček

Izhodišče: Orofacialni razcep je najpogostejša prirojena obrazna nepravilnost. Otroci z razce- pi v orofacialnem področju imajo poleg težav z govorom tudi težave na področju hranjenja in požiranja. Raziskave kažejo, da so simptomi težav s hranjenjem precej raznoliki. V skrajnih prim- erih je potrebno uvesti nadomestne načine hranjenja. V Sloveniji še ni bila narejena raziskava o pojavljanju težav s hranjenjem pri otrocih z razcepi v orofacialnem področju.

Slovenian Medical

Journal

(2)

1 Introduction

Orofacial clefts are the most common congenital craniofacial malformations. In Slovenia, about 1.77 individuals with clefts in the orofacial region are born per 1,000 live births (1). Most orofacial clefts occur as a combination of genetic and external factors and develop in the early stages of pregnancy (2).

Orofacial clefts are divided into four groups. The first group includes a cleft lip (cheiloschisis – CL) or a lip and alveolus on one or both sides (cheilognathoschisis unilateralis/bilateralis – CLA). The second group includes a cleft lip, alveolus and palate on one or both sides (cheilognathopal- atoschisis unilateralis – UCLP / bilateralis – BCLP). The third group of orofacial clefts

Metode: Ugotoviti smo želeli pogostost in vrsto težav s hranjenjem otrok z razcepi v orofacialnem področju, starih od pet mesecev do šest let. V ta namen smo izoblikovali anonimni vprašalnik za starše o vrsti orofacialnega razcepa, možnih otrokovih težavah pri hranjenju, požiranju in govo- ru ter pripomočkih za hranjenje. Vprašalnik je bil najprej pilotsko preizkušen pri starših otrok z orofacialnimi razcepi med kontrolnimi ambulantnimi pregledi, nato pa poslan 93 staršem otrok z orofacialnimi razcepi, rojenih v obdobju treh zaporednih let. Z opisno statistiko smo predstavili pogostnost pojavljanja težav ter analizirali dejavnike, ki so se pogosteje pojavljali pri otrocih s težavami pri hranjenju.

Rezultati: Starši 58 otrok z orofacialnimi razcepi so izpolnili vprašalnik. Ugotovili smo, da je ime- lo skoraj tri četrtine otrok težave z vzpostavitvijo hranjenja takoj po rojstvu, kasneje so se težave s hranjenjem zmanjšale, vendar so bile v četrtini primerov še vedno prisotne do zaključenega kirurškega zdravljenja. Največ težav se je pojavljalo zaradi uhajanja hrane in pijače skozi nos, požiranja zraka pri hranjenju po steklenički in podaljšanega časa hranjenja. Otroci z razcepom neba z razcepom ustnice ali brez so imeli bistveno več težav s hranjenjem kot otroci samo z razcepom ustnice. Večina staršev je za hranjenje uporabljala prilagojeno stekleničko in je bila z upo- rabo zadovoljna. Več kot polovica staršev je izjavila, da so prejeli premalo informacij v zvezi s hranjenjem pred in takoj po rojstvu otroka.

Zaključek: Kljub dobro organizirani multidisciplinarni skrbi za otroke z orofacialnimi razcepi v Sloveniji starši še vedno pogrešajo konkretna navodila o hranjenju otroka z orofacialnim razcepom, zaradi česar bo potrebno dodatno izobraziti predvsem medicinske sestre, ki delajo v po- rodnišnicah. Glede na visoke cene pripomočkov za hranjenje otrok z orofacialnimi razcepi bi bilo dobrodošlo, da bi kril stroške ZZZS.

Cite as/Citirajte kot: Jelenc N, Eberlinc A, Hočevar Boltežar I. Feeding and swallowing disorders in children with clefts in the orofacial region. Zdrav Vestn. 2019;88(9–10):405–14.

DOI: https://doi.org/10.6016/ZdravVestn.2892

includes cleft palate (palatoschisis – CP).

The fourth group includes rare forms of orofacial clefts in other areas of the face (2,3). Orofacial cleft may occur as part of a syndromic symptomatology. There are over 400 syndromes associated with cleft lip and/or palate (4). About 30% of all orofacial clefts occur within a syndrome (5).

A cleft in the orofacial region can great- ly affect the appearance, speech, hearing, as well as growth and development of the face, and it also psychosocially and eco- nomically affects the child and his or her family, thereby significantly reducing the quality of life (2,6).

Feeding is the basic function of every human being. Feeding and swallowing en-

(3)

compasses the whole act of taking food in- to your mouth, forming a bite and having the bite travel through the pharynx into the esophagus and stomach (7).

Numerous studies have shown that children with orofacial clefts have problems with feeding to various extents, but experts are not consistent about the symptomatology of the problems. Choking, coughing, excessive air intake and prolonged feeding time can lead to inappro- priate weight gain, dehydration and the need for alternative methods of feeding (8,9). The most commonly reported problem with feeding newborns with cleft palate are oral motor dysfunction and nasal regurgitation, as well as low feeding effi- ciency. The greatest risk of feeding problems exists in children with the Robin sequence (10).

De Vries and coworkers (8) used questionnaires for parents (90 participants) to determine the frequency of feeding problems in people with CP. As many as 67% of children with CP, who were included in the study, indicated a high risk of developing feeding problems. It was often necessary to introduce feeding using a nasogastric tube (in 32%). Jones (11) reported feeding problems in 25% of children with different orofacial clefts, and Spriestersbach with coworkers (12) found that as many as 73%

of children with isolated CP had feeding problems. Reid and coworkers (13) investigated feeding skills in infants with schisis and tried to identify the risk factors. They found that 32% of newborns with orofacial clefts have poor feeding skills, with prevalence falling to 19% at 3 months and 15% at 14 months of age. Only rare newborns with certain orofacial clefts can be breastfed. De Vries and coworkers (8) not- ed that 86% of mothers of children with cleft palate had never tried breastfeeding.

Trenouth and Campbell (14) found that only in newborns with isolated CL were they able to maintain adequate weight by breastfeeding alone.

Reid and coworkers (15) investigated the suction of newborns, born with

orofacial clefts. They found that children with isolated CL showed similar sucking as their peers, while in newborns with CP with or without CL they detected an altered pattern of sucking.

Experts point out that feeding must be treated completely differently when it comes to orofacial cleft within the craniofacial syndrome. In addition to the affected facial structures, feeding is influenced by a number of other structural, respirato- ry and neuromuscular features of the child (16,17).

It is important to recognize that the treatment of children with orofacial clefts is time-consuming and complex, and from the birth of a child on, various specialists are included, who meet regularly, together plan the treatment of the most demand- ing children with orofacial clefts, evaluate their work, look for causes of failure and try to clarify and eliminate them (18).

Team members identify the arisen disorders, treat the child with an orofacial cleft themselves, or refer them to other professionals with narrower expertise, close to the child’s home (19,20).

To date, there has not yet been a study in Slovenia that would systematically investigate feeding and swallowing problems in children with orofacial clefts and their resolution.

2 Methods

The study population was preschool children with a cleft in the orofacial region, aged 6 months to 6 years. For the purpose of the research, an anonymous questionnaire was developed based on the literature data (17,21). The questions were related to general information about the child, information about his or her feeding and swallowing, the use of feeding gadgets, accompanying problems besides feeding (sensitivity to being touched in the mouth and teeth being brushed, difficulty developing speech, parental stress during feeding), and data on the support and assistance of practitioners in feeding a child

(4)

with orofacial cleft.

The questionnaire was first pilot tested with 19 parents who brought their children with orofacial clefts for examina- tion to a maxillofacial surgeon, and then distributed to all parents of children with orofacial clefts born in three consecutive years, from 1 Jan. 2013 to 31. Dec. 2016.

The questionnaire was anonymous. Only with the added name of the child and the signed permission of the parents could we summarize the child’s medical data from the medical records and complete the results of the questionnaire (gestational age at birth and type of orofacial cleft).

The results were analyzed with Excel 2013 and IBM SPSS Statistics 22.0. We presented them with descriptive statistics, and then compared the results of the questionnaire between a group of children with orofacial clefts who had problems with feeding and swallowing, and a group of children for whom parents clamed they had no feeding problems. We used the chi- square test or Fisher’s exact test.

The study was approved by the Medical Ethics Committee of the Republic of Slo- venia (Decision No. 0120–107/2017).

3 Results

The response to the sent questionnaire

was 47%, of the 93 sent questionnaires 44 were sent back to us. A total of 58 questionnaires were included in the final analysis, besides the 44 returned questionnaires there were also 14 fully completed ones from the pilot survey, namely, those completed by the parents of children 6 years and older. The final group consisted of children between the ages of six and 72 months (mean 30 months, standard devi- ation 16.8 months). The study participants were divided into groups according to the type of orofacial cleft (Table 1).

A total of 53 (91.4%) children surveyed were born after the 37th week of gestational age. In ten children (17.2%), schisis ap- peared within a syndrome: in 6 children as a part of Robin sequence, in 2 as a part of van der Woude syndrome, 1 had orofaciodigital syndrome and 1 had Down’s syndrome in addition to the orofacial cleft syndrome.

Problems with feeding were experienced by 42 (72.4%) children at some point in their lives. In 24 children (40.4%) problems still persisted one month after birth, while 14 children (24.1%) had problems until the end of surgical treatment, and 8 (13.8%) had problems all the time.

Among these 8 children three had CP, four had UCLP, and one had bilateral CL.

Two had an orofacial cleft that was part of a syndrome (Robin sequence, van der Woude syndrome).

Eleven children were breastfed for at least one month, among them were eight children with CL and with or without al- veolar cleft and 3 children with CP (one had submucosal cleft palate and one in- complete soft palate cleft).

In a certain period there were 6 children with orofacial cleft being fed through a nasogastric tube. The average feeding time through a nasogastric tube was 3.2 months, and all children had the tube inserted in the first month of their lives. Of the children fed through the tube, three had CP and three had UCLP. Four of them had CLP within a syndrome. Two had Robin sequence, one had van der Woude Table 1: Type of orofacial cleft by gender of participants in the study

(N = 58).

Legend: UCL - unilateral cleft lip, UCLA - unilateral cleft lip and alveolus, BCLA - bilateral cleft lip and alveolus, CP - cleft palate, UCLP - unilateral cleft lip, alveolus and palate, BCLP - bilateral cleft lip, alveolus and palate.

Type Men (%) Women (%) Total (%)

UCL 3 (5.2) 2 (3.4) 5 (8.6)

UCLA 1 (1.7) 2 (3.4) 3 (5.2)

BCLA 1 (1.7) 2 (3.4) 3 (5.2)

CP 11 (19.0) 13 (22.4) 24 (41.4)

UCLP 9 (15.5) 7 (12.1) 16 (27.6)

BCLP 6 (10.3) 1 (1.7) 7 (12.1)

Total 31 (53.4) 27 (46.6) 58 (100.0)

(5)

syndrome and one had orofaciodigital syndrome. Only one child was fed through a gastrostomy tube, namely, from 8 to 16 months of age, and had had a nasogastric tube inserted before that. In addition to CP, he also had Robin sequence, he was a twin and a premature baby.

Table 2 shows the feeding and swallowing problems of children with orofacial clefts by frequency:

In addition to the stated feeding problems, 20 children with orofacial clefts had poor tolerance of mouth contact, and 14 children had difficulty having their teeth brushed. Three of the six children fed through a nasogastric tube also had problems with being touched in the mouth, while these problems were only present in one third of the children, who didn’t use the tube. Of the 14 children who had problems with biting and chewing, 9 poorly tolerated touching of the mouth, 2 were fed through a nasogastric tube for

a certain period of time, 1 of these 2 was then fed through a gastrostomy tube for another 8 months.

In our sample, according to parents, 24 children had speech problems, 11 of whom had CP and 13 had CLP. In 8 children, speech developed normally without the help of a speech therapist. Among them, five had CL or CLA, three had CP and two had UCLP. 24 children were too young for their parents to consider the presence of speech problems.

The use of a modified bottle helped to reduce feeding problems in 35 children (81.4% of those who used a modified bottle). After they started to use the palatal obturator, parents of 19 children (75% of those who used the palatal obturator) noticed that feeding problems also decreased.

More than a third of parents (20 people) experienced feeding their child in a certain period of time as stressful.

Figure 1 shows which experts advised parents about feeding and swallowing.

Only 15 (25.9%) parents had known before birth that they would give birth to a child with an orofacial cleft. They all had at least a cleft lip and alveolus. Among them there were no children with only CL and no children with only CP.

A good two-thirds (41 parents) would have liked to attend education regarding feeding and swallowing even before or immediately after the birth of a child with an orofacial cleft.

When comparing children with orofacial clefts who have feeding problems at some point in their lives and children without problems, we found a statistically significant difference between the groups regarding the type of orofacial cleft, the presence of a syndrome, subsequent speech problems, and parents experienc- ing stress when feeding the child. A bor- derline characteristic was also weight gain (Table 3). Due to the small number of children who have had feeding problems all the time, we have only shown the results of the comparison of these children to others with descriptive statistics (Table 4).

Table 2: Procedural characteristics.

Feeding problems Number (%)

Nasal regurgitation before surgical treatment is completed 37 (63.8) Excessive air intake while bottle feeding 25 (43.1) The child falls asleep while being bottle fed 25 (43.1)

Prolonged feeding time 23 (39.7)

Food escaping the mouth while bottle feeding 20 (24.5)

Problems with biting and chewing 14 (24.6)

Solid food escaping the mouth 14 (24.1)

Coughing, choking while eating solid food 12 (20.7) Vomiting, dribbling while bottle feeding 12 (20.7)

Crying while bottle feeding 12 (20.7)

Coughing, choking while bottle feeding 11 (19.0)

Improper weight gain 9 (15.5)

Crying while eating solid food 7 (12.1)

Feeding through a tube 6 (10.3)

Vomiting while eating solid food 3 (5.2)

Nasal regurgitation after surgical treatment is completed 1 (1.7)

(6)

4 Discussion

Given that between 30 and 40 children with orofacial clefts are born in Slovenia per year (1), we included in our sample a fairly large proportion of children (58 per- sons) aged between 6 months and 6 years.

We decided to send a questionnaire to children who were 6 months to 3 years old at the time of the survey because we want- ed to get information primarily about the initial feeding problems. In the pilot part of the survey to evaluate the questionnaire, we covered children who were regularly examined by a maxillofacial surgeon. We then included only pre-school children in the analysis, as extending the period from the birth of the child to completing the questionnaire may cause some problems to fade from their parents’ memories.

Reid and coworkers (13) investigated the incidence of feeding problems in children with orofacial clefts and found that the problems decreased with age and the incidence of feeding problems decreased by the age of 14 months to 15%. The re- Table 3: Comparison of the children with orofacial clefts regarding feeding problems some time in life (N = 58).

Legend: CL - cleft lip, CLA – cleft lip and alveolus, CP – cleft palate, CLP – cleft lip, alveolus and palate.

Feeding problems

(some time in life) p N = 42YES NO

N = 16

CL / CLA (N = 11) 3 8 0.001

CP / CLP (N = 47) 39 3 0.001

Presence of syndrome (N = 10) 10 0 0.048

Problems with speech (N = 24) 20 4 0.034

Nasogastric feeding (N = 6) 6 0 0.173

Improper weight gain (N = 9) 9 0 0.051

Poor tolerance of mouth contact

(N = 20) 16 4 0.371

Parents experience stress during

feeding (N = 20) 18 2 0.032

Table 4: Comparison of groups of children with orofacial clefts regarding persistant feeding problems (N = 58).

Feeding problems (present all time) N = 8YES NO N = 50

CL / CLA (N = 11) 1 10

CP / CLP (N = 47) 7 40

Presence of syndrome (N = 10) 2 8

Problems with speech (N = 24) 2 22

Nasogastric feeding (N = 6) 3 3

Improper weight gain (N = 9) 2 7

Poor tolerance of mouth contact (N = 20) 4 16 Parents experience stress while feeding (N = 20) 6 14

Figure 1: The source of information about feeding of the child with orofacial cleft.

0 5 10 15 20 25 30

sults of our study showed that in the first 35

month of life, less than three-quarters of children had feeding problems, the problems persisted at 24% until the comple- tion of surgical treatment. This means that most feeding problems occurred when trying to breastfeed or when bottle feeding during the first months of life, only in a small number of cases did problems persist later on introducing solid and formed foods. The findings of Jones and coworkers (11) as well as Reid (10) were similar.

There is generally little information on the frequency of swallowing problems in the population of children. The U.S. reports that in 2012, about 1% of the child population in the country had difficulty swallowing, but only about 13% sought appropriate professional help (22). There is no data for Slovenia, however, when comparing the incidence of dysphagia in the American study among the general child population and the results of our study, it is clear that feeding problems are a significant problem for children with orofacial clefts even after surgical treatment is completed. Only two of eight children (13.8%) who had feeding problems after completing surgical treatment had a syndrome. Swallowing problems are therefore likely not only due to different anatomical conditions (orofacial cleft) but also due to other factors.

Peterson-Falzone notes that a large proportion of children with orofacial clefts have problems with establishing a normal feeding pattern during the early period, the most problems arising due to communication between the oral and nasal cavities. This results in an inability to create negative pressure in the oral cavity.

This reduces the suction capability (3). It was the nasal regurgitation that was the most common problem in our study, as well as excessive air intake while bottle feeding, drowsiness during bottle feeding, prolonged feeding time, food escaping from the mouth while bottle feeding, and problems with biting and chewing. Most problems were therefore due to communi-

(7)

4 Discussion

Given that between 30 and 40 children with orofacial clefts are born in Slovenia per year (1), we included in our sample a fairly large proportion of children (58 per- sons) aged between 6 months and 6 years.

We decided to send a questionnaire to children who were 6 months to 3 years old at the time of the survey because we want- ed to get information primarily about the initial feeding problems. In the pilot part of the survey to evaluate the questionnaire, we covered children who were regularly examined by a maxillofacial surgeon. We then included only pre-school children in the analysis, as extending the period from the birth of the child to completing the questionnaire may cause some problems to fade from their parents’ memories.

Reid and coworkers (13) investigated the incidence of feeding problems in children with orofacial clefts and found that the problems decreased with age and the incidence of feeding problems decreased by the age of 14 months to 15%. The re- Table 3: Comparison of the children with orofacial clefts regarding feeding problems some time in life (N = 58).

Feeding problems

(some time in life) p N = 42YES NO

N = 16

CL / CLA (N = 11) 3 8 0.001

CP / CLP (N = 47) 39 3 0.001

Presence of syndrome (N = 10) 10 0 0.048

Problems with speech (N = 24) 20 4 0.034

Nasogastric feeding (N = 6) 6 0 0.173

Improper weight gain (N = 9) 9 0 0.051

Poor tolerance of mouth contact

(N = 20) 16 4 0.371

Parents experience stress during

feeding (N = 20) 18 2 0.032

Table 4: Comparison of groups of children with orofacial clefts regarding persistant feeding problems (N = 58).

Feeding problems (present all time) N = 8YES NO

N = 50

CL / CLA (N = 11) 1 10

CP / CLP (N = 47) 7 40

Presence of syndrome (N = 10) 2 8

Problems with speech (N = 24) 2 22

Nasogastric feeding (N = 6) 3 3

Improper weight gain (N = 9) 2 7

Poor tolerance of mouth contact (N = 20) 4 16 Parents experience stress while feeding (N = 20) 6 14

Figure 1: The source of information about feeding of the child with orofacial cleft.

0 5 10 15 20 25 30 35

cation between the oral and nasal cavities.

Similar findings have been made by other authors as well (8,13,21). Drowsiness during feeding could be attributed to the

higher energy consumption of the child in the case of unsuccessful sucking, which sooner leads to fatigue during feeding.

Subsequent biting and chewing problems could be explained, at least in part, by altered sensibility in the oral cavity due to orofacial clefting and the consequences of treatment, as nine out of 14 children who had biting and chewing problems for at least a certain period of time had poor tolerance of mouth contact.

McDonald found in her study that the type of orofacial cleft had a significant effect on breastfeeding, for 60% of infants with CL were breastfed for more than 6 months, while only few infants with CP were breastfed for more than 6 months (21). In our study, only eleven infants with orofacial clefts were breastfed (19%), in- cluding three with CP, which is in line with the data in professional literature (21,14).

We have found that the type of orofacial cleft has a statistically significant effect on feeding and swallowing problems, since children with CL or CLA have significantly less problems than children with CP or CLP (Table 3). This result is a logical con-

(8)

sequence of the anatomical conditions that exist until the end of surgical treatment, rarely even later. According to literature data, children with feeding problems at some point in life were more likely to gain weight inappropriately, were fed through a nasogastric tube, and had a syndrome in addition to orofacial cleft (Table 3).

In 3 out of 6 children fed through a nasogastric tube, parents described poor tolerance of oral cavity contact. These children are most likely to have developed excessive sensitivity due to their lack of experience at an early age when neural connections between the periphery and the central nervous system are still being established and matured.

Only in 8 children did feeding problems persist even after surgical treatment.

This group of children was also often fed by means of alternative methods (3/8), and parents of 6 children experienced stress during feeding. In addition to the possible suboptimal anatomical conditions for feeding and swallowing, there were most likely other factors involved - e.g. altered experience of sensory stimuli as a result of developments in the oral cavity during the first years of life or other accompanying problems. As many as half of the children with persistent feeding problems had poor tolerance of oral cavity contact. The per- ception of stimuli in the oral cavity is also affected by surgery, the use of a nasogastric tube or gastrostomy tube for feeding, and to some extent the use of a palatal obturator (6,23).

Children who had feeding problems were more likely to gain weight improper- ly and needed an alternative feeding method (Table 3). The introduction of a nasogastric tube or even a gastrostomy tube for feeding is the consequence of inadequate growth and development of the child. In the vast majority of children, the alternative method of feeding lasted less than a year, and then they were again fed through the mouth, even though as many as five children had a cleft palate. One infant with CP within the Robin sequence was first fed

through a nasogastric tube and then fed for eight months through a gastrostomy tube. The boy was one of twins, born more than a month premature, which could also be a cause of feeding problems at least immediately after birth. Among the 6 children who needed an alternative method of feeding, four had orofacial cleft within a syndrome. Even in the professional literature, it is reported that an alternative feeding method should be more frequent- ly introduced in children with syndromes than in those with an orofacial cleft, that is not part of a syndrome (16,17).

To ensure that children with orofacial clefts can be fed successfully, there are cus- tomized bottles that increase food intake while preventing the child from swallowing too much air (23). According to parents’ reports, we found that there are few types of custom bottles available in Slove- nia. At the maternity hospital, parents are usually offered one brand of bottle, which is not suitable for all children, and pres- ents a major financial burden for parents.

Palatal obturator was used by 26 children. Although the palatal obturator is part of preoperative jaw orthopedic treatment (2), some studies indicate that its use in combination with other measures is one of effective methods in establishing effective feeding of a baby with schisis (20). In our study, parents noticed a decrease in feeding problems after the palatal obturator was used in as many as three quarters of children who received a palatal obturator as part of preoperative preparation for orofacial cleft treatment.

Due to the procedures of successful early diagnosis, some parents are aware of orofacial cleft before the child is born.

In our study, there were about a quarter of such parents (15 people). It would be wise to involve a professional team to help expectant parents even before the child is born, when the parents find out that their child would be born with schisis. Each of the experts would thus provide them with information about the functional disorders that he or she mostly deals with and

(9)

how to solve these problems.

It would also be welcome to have a clinical speech therapist, in addition to the maxillofacial surgeon, see the children and their mothers in maternity hospitals immediately after birth, who would provide parents with information regarding the relationship between feeding and speech development and would further monitor the child. In every maternity hospital, at least one nurse or other health professional should be specifically trained in functional disorders of children with orofacial clefts and feeding assistance in order for parents to receive immediate help and to reduce their distress when unable to feed a newborn with orofacial cleft.

In their written comments, the parents emphasized the problem of inadequate support by nurses and midwives in feeding. It would be reasonable to build on the research by examining the knowledge of medical staff about working with children with orofacial clefts, in order to find out in which parts of Slovenia the professional competence of the staff is the worst. Cur- rently, education activities on the topic of feeding and swallowing of children with orofacial clefts in the orofacial area are already taking place in Slovenia, where professionals receive knowledge of basic

ways of working, counseling and selecting feeding gadgets. In the future, it might make sense to investigate the link between feeding and speech and the impact of decreased oral activity on speech and lan- guage development.

5 Conclusion

Feeding difficulties in children with orofacial clefts are a common occurrence.

At some point in their lives, less than three-quarters of the children taking part in the study had feeding problems, with a good seventh having problems persisting even after the completed surgical treatment. The aim of the study was to empha- size the importance of having the child treated early by a multidisciplinary team that could prevent many problems arising from prolonged inefficient feeding. It is important to bear in mind that feeding difficulties have a great impact on the whole family, as feeding not only satisfies the need for calorie intake but also strength- ens the bond between mother and child.

It can be concluded that the treatment of children with orofacial clefts in Slovenia is good, but early counseling on feeding and selecting appropriate feeding techniques and gadgets are insufficient.

References

1. Koželj V. Računalniški informacijski sistem za spremljanje orofacialnih shiz v Sloveniji kot vir za epidemiološko analizo obdobij 1973-1993 in 1993-2012. Zobozdrav Vestn. 2015;70(3-5):135-42.

2. Watkins SE, Meyer RE, Strauss RP, Aylsworth AS. Classification, epidemiology, and genetics of orofacial clefts. Clin Plast Surg. 2014;41(2):149-63. DOI: 10.1016/j.cps.2013.12.003 PMID: 24607185

3. Peterson-Falzone SJ, Trost-Cardamone JE, Karnell MP, Hardin-Jones M. The Clinician’s Guide to Treating Cleft Palate Speech. Philadelphia: Mosby Elsevier; 2006.

4. Shkoukani MA, Lawrence LA, Liebertz DJ, Svider PF. Cleft palate: a clinical review. Birth Defects Res C Embryo Today. 2014;102(4):333-42. DOI: 10.1002/bdrc.21083 PMID: 25504820

5. Crowley L, Gist KL, Gregson KM, Hufnagle K, Peterson-Falzone S, Smythe NC, et al. Your baby’s first year.

Chapel Hill: CPF Publications Committee; 2012.

6. Eberlinc A. Estetski problem pri pacientih s heilognatopalatoshizo. In: Zbornik prispevkov: sodobne metode in nove tehnologije v maksilofacialni kirurgiji in stomatologiji; 29. november 2014; Brdo pri Kranju, Sloveni-ja. Ljubljana: Združenje za maksilofacialno in oralno kirurgijo Slovenije; 2014. p. 75

7. Murry T, Carrau RL. Clinical management of swallowing disorders. 2nd ed. San Diego: Plural Publishing;

2006.

8. de Vries IA, Breugem CC, van der Heul AM, Eijkemans MJ, Kon M, Mink van der Molen AB. Prevalence of feeding disorders in children with cleft palate only: a retrospective study. Clin Oral Investig.

2014;18(5):1507-15. PMID: 24122307

(10)

9. Perlman AL, Christensen J. Topography and functional anatomy of the swallowing structures. In: Perlman AL, Schulze-Delrieu K, eds. Deglutition and its disorders. San Diego: Singular Publishing Group, INC; 1997.

pp. 15-42.

10. Reid J. A review of feeding interventions for infants with cleft palate. Cleft Palate Craniofac J.

2004;41(3):268-78. DOI: 10.1597/02-148.1 PMID: 15151444

11. Jones WB. Weight gain and feeding in the neonate with cleft: a three-center study. Cleft Palate J.

1988;25(4):379-84. PMID: 3060285

12. Spriestersbach DC, Dickson DR, Fraser FC, Horowitz SL, McWilliams BJ, Paradise JL, et al. Clinical research in cleft lip and cleft palate: the state of the art. Cleft Palate J. 1973;10:113-65. PMID: 4220387

13. Reid J, Kilpatrick N, Reilly S. A prospective, longitudinal study of feeding skills in a cohort of babies with cleft conditions. Cleft Palate Craniofac J. 2006;43(6):702-9. DOI: 10.1597/05-172 PMID: 17105331

14. Trenouth MJ, Campbell AN. Questionnaire evaluation of feeding methods for cleft lip and palate neonates.

Int J Paediatr Dent. 1996;6(4):241-4. DOI: 10.1111/j.1365-263X.1996.tb00252.x PMID: 9161191

15. Reid J, Reilly S, Kilpatrick N. Sucking performance of babies with cleft conditions. Cleft Palate Craniofac J.

2007;44(3):312-20. DOI: 10.1597/05-173 PMID: 17477747

16. Miller CK. Feeding issues and interventions in infants and children with clefts and craniofacial syndromes.

Semin Speech Lang. 2011;32(2):115-26. DOI: 10.1055/s-0031-1277714 PMID: 21948638

17. Paes EC, de Vries IA, Penris WM, Hanny KH, Lavrijsen SW, van Leerdam EK, et al. Growth and prevalence of feeding difficulties in children with Robin sequence: a retrospective cohort study. Clin Oral Investig.

2017;21(6):2063-76. DOI: 10.1007/s00784-016-1996-8 PMID: 27868158

18. Hodgkinson DP, Brown S, Duncan D, Grant C, McNaughton A, Thomas P, et al. Management of children with cleft lip and palate: A review describing the application of multidisciplinary team working in this condition based upon the experiences of a regional cleft lip and palate centre in the United Kingdom. Fetal Matern Med Rev. 2005;16(1):1-27. DOI: 10.1017/S0965539505001452

19. Jarc A, Hočevar Boltežar I. Skrb za osebe z razcepom ustnic, čeljusti in neba v Sloveniji. Družbena skrb za vzgojo, izobraževanje in socialno varstvo oseb s posebnimi potrebami - deset let kasneje : zbornik referatov. 2000 Oct 18-20; Portorž, Slovenija. Ljubljana: Društvo defektologov Slovenije; 2000.

20. Koželj V, Drevenšek M, Hočevar Boltežar I, Jarc A. Koncept zdravljenja orofacialnih shiz. Sodobni kirurško ortodontski koncepti : [zbornik predavanj]. 12. Čelešnikovi dnevi. 2006 Nov 25; Ljubljana, Slovenija.

Ljubljana: Združenje za maksilofacialno in oralno kirurgijo Slovenije; 2000.

21. McDonald SH. To Investigate Infant Feeding in Children born with a Cleft in the West of Scotland. [[Thesis]].

Glasgow: Faculty of medicine; 2008.

22. Bhattacharyya N. The prevalence of pediatric voice and swallowing problems in the United States.

Laryngoscope. 2015;125(3):746-50. DOI: 10.1002/lary.24931 PMID: 25220824

23. Devi ES, Sai Sankar AJ, Manoj Kumar MG, Sujatha B. Maiden morsel - feeding in cleft lip and palate infants.

J Int Soc Prev Community Dent. 2012;2(2):31-7. DOI: 10.4103/2231-0762.109350 PMID: 24478964