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International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e77

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International Journal of Pediatric Otorhinolaryngology

journal homepage: http: / /www.i jporlonl ine.com/

Treatment outcome after neonatal cleft lip repair in 5-year-oldchildren with unilateral cleft lip and palate

Olga Ko�skov�a a, b, d, *, Jitka Vokurkov�a a, b, d, Jan Vokurka c, d, Alena Bry�sova c, d,Pavel �Senovský c, d, Julie �Cefelínov�a a, Darina Luk�a�sov�a a, Petra Dorociakov�a e,Juraj Abelovský d

a Department of Pediatric Plastic Surgery - Department of Pediatric Surgery, Orthopedics and Traumatology, University Hospital Brno, �Cernopolní 9, 613 00Brno, Czech Republicb Department of Burns and Reconstructive Surgery, University Hospital Brno, Jihlavsk�a 20, 625 00 Brno, Czech Republicc Clinic of Dentistry, St. Anne's Faculty Hospital, Peka�rsk�a 53, 656 91 Brno, Czech Republicd Faculty of Medicine, Masaryk University, Brno, Czech Republice Recetox, Faculty of Science, Masaryk University, Kamenice 753/5, 625 00, Brno, Czech Republic

a r t i c l e i n f o

Article history:Received 9 March 2016Received in revised form10 May 2016Accepted 12 May 2016Available online 26 May 2016

Keywords:Unilateral cleft lip and palateNeonatal cleft lip surgeryOne-stage palatal repairThe GOSLON YardstickSpeech outcomes measurements

* Corresponding author. Department of PediatriTraumatology, University Hospital Brno, �Cernopolní 9,

E-mail address: koskova.olga@gmail.com (O. Ko�sk

http://dx.doi.org/10.1016/j.ijporl.2016.05.0240165-5876/© 2016 Elsevier Ireland Ltd. All rights rese

a b s t r a c t

Introduction: The aim of this study was to assess speech outcomes and dental arch relationship of 5-year-old Czech patients with unilateral cleft lip and palate (UCLP) who have undergone neonatal cleft liprepair and one-stage palatal closure.Methods and materials: Twenty-three patients with UCLP, born between 2009 and 2010, were includedin the study. Three universal speech parameters (hypernasality, articulation and speech intelligibility)have been devised for speech recordings evaluation. Outcomes of dental arch relationship were evalu-ated by applying the GOSLON Yardstick and subsequently compared with the GOSLON outcome of othercleft centers.Results: Moderate hypernasality was present in most cases, the mean value for articulation and speechintelligibility was 2.07 and 1.93, respectively. The Kappa values for inter-examiner agreement for all thethree speech outcomes ranged from 0.786 to 0.808. Sixty-three percent of patients were scored GOSLON1 and 2, 26% GOSLON 3, and 10% GOSLON 4. GOSLONmean score was 2.35. Interrater agreement was verygood, represented by kappa value of 0.867.Conclusion: The treatment protocol, involving neonatal cleft lip repair and one-stage palatal repairperformed up to the first year of UCLP patient's life, has shown good speech outcomes and produced verygood treatment results in regard to maxillary growth, comparable with other cleft centers.

© 2016 Elsevier Ireland Ltd. All rights reserved.

1. Introduction

The incidence of cleft lip and palate (CLP), one of the mostcommon craniofacial malformations, is 11.13 for 10 000 live birthsin the Czech Republic [1]. Treatment protocol of CLP is different ineach cleft center. In the Czech Republic there is an early surgerytrend being observed [2,3]. In our department cleft lip repair isperformed in neonatal period and cleft palate repair begins at theage of 6 months and later respectively.

c Surgery, Orthopedics and613 00 Brno, Czech Republic.ov�a).

rved.

Final treatment outcomes of CLP patients cannot be fullyassessed until adulthood; however, there are some predeterminedperiodic follow-up visits during the CLP patient's maturing. Ac-cording to Eurocleft study [4] patients with CLP should undergofollow-up assessment sessions at 5, 10 and 18 years of age whichinclude standardized speech audio recordings, photos and dentalmodels. Inter-center comparison is necessary for treatment successrating.

The purpose of the study was to analyze speech outcomes anddental arch relationship in the same sample group of 5-year-oldchildren with unilateral cleft lip and palate (UCLP), treated withneonatal cleft lip repair and one-stage palatal reconstruction.

There is no standard speech-sampling protocol or guidelines forcapturing cleft-palate speech errors in Czech language which can

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e7772

be used for inter-language comparison. Speech-sampling protocolscreated by Kerekretiova [5] or Brohm [6] are mostly used for CzechCLP patients but, in our view, they are either too extensive or tooshort and thus insufficient. Therefore the study was focused oncreating a contemporary speech protocol, specifically designed forCzech language, and its assessment in accordance with the recog-nized speech therapy schemes currently available.

There are more options for assessment of maxilla's growth andresulting analysis of the occlusal effects of primary surgery [7]. TheGreat Ormond Street, London and Oslo, Norway Yardstick, morecommonly known as the GOSLON Yardstick [8], was applied in thestudy. The outcomes evaluated by GOSLON Yardstick werecompared with other cleft center results.

2. Material and methods

The study was based on the approval of the ethics committee ofSt. Anne's University Hospital in Brno (reference number 25V/2015).

2.1. Subjects

In the Department of Pediatric Plastic Surgery, the UniversityHospital Brno, 25 patients, born between January 2009 andDecember 2010, were treated. Two patients were excluded becauseof diagnosis of syndrome associated with cleft; all the remainingchildren proceeded in the study. All patients were operated on byan experienced surgeon (Vokurkova, MD). Speech audio recordings,photo documentation and dental models were being completed inall the children with UCLP. Children's age at the time of lip andpalate closure, gender and presence of oro-nasal communicationwere also observed. The mean age of the 23 patients was 5.3 years(4.5e6.2). Unilateral cleft lip and palate affected the left side in 70%of the children in the study. Seventeen patients (77%) were boys.The mean age at cleft lip surgery was 6.2 days (2e18 days), themean age at palate repair was 6.9 months (6e12months). Four outof the 23 children had an oro-nasal fistula after primary palatalsurgery.

2.2. Surgical procedures and treatment protocol

The optimal surgery timing is determined by each cleft center[4,9,10]. In our department, cleft lip repair is performed in neonatalperiod (0e28 days of age). Amodified Fisher's method, combining acorrection of the lip and nose [11], was used in each case in thestudy. Early lip closure, as a natural nasoalveolar molding, helpsbring both alveolar segments closer [2]. All patients underwentpalate closure between six and twelve months of age. One stagepalate repair using intravelar veloplasty is preferred. The emphasisis on the most proper restoring of all abnormal muscle insertions[12], minimal lateral incisions, and no fracture of hamulus. A vomerflap was not applied as a standard; it was used only in a few uniquecases of particularly serious clefts. In case of velopharyngealinsufficiency presence at pre-school age, the Furlow palatoplasty or

Table 1Speech outcomes evaluation e hypernasality, articulation and speech intelligibility.

Score Hypernasality Articulation

1 Absent Normal articulation or other com2 Minimal Weak pressure consonants, prese3 Mild Mild cleft type compensatory mis4 Moderate Moderate cleft type compensator5 Severe Severe cleft type compensatory m

the veloplasty with ‘‘butterfly-suture’’, based on principles byHaase's method, is performed [13].

Every patient comes for follow-ups according to Eurocleft study[4]. Speech development is observed after palate repair and earlyspeech therapy is indicated. We consider the follow up at the age offive as one of the most crucial ones. Speech progress assessment isdetermined by standardized speech audio recordings. Dentalmodels are made for exploration of dental arch relationship for thepurpose of planning the future orthodontics therapy beforeattending school.

2.3. Assessment

2.3.1. Speech recordingsA new system for evaluating speech outcomes in CLP patients

was created for Czech language. At first, it was necessary to build aset of speech tests. Its scheme was based on principles developedby Kerekr�etiov�a [5], then it was revised and edited in accordancewith Lohmander et al. [14]. The final speech protocol consists ofarticulation test (based on description of sample of pictures), testspecialized on repetition of sentences containing high-pressuresensitive consonants and nasal consonants, counting from 1 to 10,connected speech e re-telling of a previously told story. Hyper-nasality was analyzed also by Gutzmann test and Czermak's Mirrortest. All children speech samples were recorded in the same roomusing the same recorder (Olympus linear PCM recorder LS-11EU).Subsequently, all audio recordings were taken for perceptualanalysis. The speech outcomes were evaluated in three categories:hypernasality, articulation and speech intelligibility. All categorieswere rated on the 5-point scale (Table 1). The evaluation of re-cordings was performed independently by two speech therapistsexperienced in treating cleft patients. Speech samples were blindedbefore perceptual evaluation. The following rules had beenrespected; speech therapists were allowed to listen to all re-cordings repeatedly before making a final assessment, but theactual rating had to be donewith no interruption. Two patients hadto be excluded from the study because of diagnosis of selectivemutism and dysphasia; twenty-one recordings were assessed.

2.3.2. The GOSLON YardstickThe Goslon Yardstick is a 5-point scoring system for evaluation

maxilla's growth and dental arch constriction which categorizes allpatient outcomes into 5 groups: from 1- excellent to 5 e very pooroutcome (Table 2). All the 23 patients included in the study wererated. None of the patients had undergone active orthodontictreatment prior to this. Two experienced orthodontists (A.B. andP.S.), who rated the models, adhered to the following rules. Prior tothe actual evaluation, each examiner was thoroughly familiarizedwith applying the GOSLON Yardstick in order to prevent systematicbias. Therefore, as the first step, testing assessment and subsequentstandardization of evaluation were performed. The final evaluationprocess, determined after a certain period, was performed by eachexaminer separately and with no interruption during the evalua-tion. The results were compared with outcomes of other cleft

Speech intelligibility

mon dysarticulation Normal (100%)nce of nasality Good (90e99%)articulation Lower (75e89%)y misarticulation Very low (50e74%)isarticulation, consonantless speech Unintelligible (49% and less)

Table 3Speech outcomes.

Score Hypernasality Articulation Speech_intelligibility

Examiner 1 Examiner 2 Examiner 1 Examiner 2 Examiner 1 Examiner 2

1 4 (19.05%) 3 (14.29%) 6 (28.57%) 5 (23.81%) 11 (52.38%) 9 (42.86%)2 4 (19.05%) 4 (19.05%) 9 (42.86%) 9 (42.86%) 3 (14.29%) 5 (23.81%)3 8 (38.10%) 8 (38.10%) 6 (28.57%) 6 (28.57%) 6 (28.57%) 5 (23.81%)4 4 (19.05%) 5 (23.81%) 0 (0.00%) 1 (4.76%) 1 (4.76%) 2 (9.52%)5 1 (4.76%) 1 (4.76%) 0 (0.00%) 0 (0.00%) 0 (0.00%) 0 (0.00%)Mean 3 2 2Average 2.79 2.07 1.93

Kappa value Standard error Kappa value Standard error Kappa value Standard error0.808 0.103 0.786 0.110 0.786 0.112

Table 2The GOSLON Yardstick (Mars et al., 2006).

Groupno.

Description of the group Outcome

1 Positive overjet with average inclined or tetroctined incisors with no crossbite or open bite. Excellent2 Positive overjet with average inclined or proclined incisors with unilateral crossbite or crossbite tendency with or without open-bite tendency around

cleft site.Good

3 Edge-to-edge bite with average inclined or proclined incisors or reverse overjet with retroclined incisors. Unilateral crossbite with or without open-bite tendency around cleft site.

Fair

4 Reverse overjet with average inclined oi- proclined incisors. Unilateral crossbite with or without bilateral crossbite tendency with or without open-bite tendency around cleft site.

Poor

5 Reverse overjet with proclined incisors, bilateral crossbite, and poor maxillary arch form and palatal vault anatomy. Verypoor

Fig. 1. 5-year-old girl with UCLP (front view)- GOSLON score 1.

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e77 73

centers worldwide. Only the studies concerning 4e7 year-old pa-tients were selected following the age categories by Nollet [15].

2.4. Statistical analysis and inter-examiner agreement

The kappa statistic was used to evaluate inter-rater reliability forboth, the speech therapists and the orthodontists. Significancelevels were set at p � 0.05. According to Landis and Koch (1977)[16], kappa value > 0.6 represents ‘‘good’’ strength of agreement,and kappa >0.8 indicates ‘‘very good’’ strength of agreement. Theanalysis was carried out using the statistical package SPSS Ver. 22.

3. Results

Speech recordings were evaluated in three categories e hyper-nasality, articulation and speech intelligibility - each rated on the 5-point scale scoring system. Mean value of hypernasality was 3(average 2.79), 38.2% patients were rated hypernasality of 3 by bothspeech therapists. Mean value of other categories was 2 (average2.07 for articulation and 1.93 for speech intelligibility). More than90% achieved rating 1 to 3 for articulation as well as speech intel-ligibility. All values are shown in Table 3. The Kappa values for inter-examiner agreement for all the three speech outcomes ranged from0.786 to 0.808, indicating good/very good agreement.

Out of the 23 patients, 13% were scored GOSLON 1 (Figs. 1e4),GOSLON 2 represented approximately 50% of children and theremaining ones were classified into GOSLON score of 3 and 4(Figs. 5e8). No dental study model was found to be in group 5.GOSLON mean score was 2.35. Inter-rater agreement was verygood, represented by kappa value of 0.867 (standard error 0.088)(Table 4).

4. Discussion

The success of the surgery depends on quality of soft tissues,severity of the cleft [17], surgeon's experience, smooth post-operative course, and highly specialized multidisciplinary

treatment from birth to early adulthood [18]. Treatment protocoland particularly timing of surgeries in cleft patients are still a highlydebated topic in academic circles. Timing of cleft lip and palatesurgical repairs is determined by each institution and country

Fig. 2. 5-year.old girl with UCLP (side face)- GOSLON score 1.

Fig. 4. 5-year-old girl with UCLP (occlusion-side view)- GOSLON score 1.

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e7774

[4,19]. In the Czech Republic there is an early surgery trend beingobserved, cleft lip repair is performed in neonatal period and cleftpalate repair follows at about 6 months of age respectively [2,3].Early cleft lip closure is considered controversial but sometimescriticized unsubstantially. It is not uncommon to find both, oppo-nents as well as supporters of this method. The first mention ofneonatal cleft lip surgery is dated to the sixties of the last century[20]. In the other decades NS Desai was an important personality inpromoting this method in England [21,22]. Early cleft lip repair isnow performed in France, the Czech Republic and Slovakia

Fig. 3. 5-year-old girl with UCLP (occlusion-frontal view) e GOSLON score 1.

[2,3,23,24]. This method's expansion has always been limited bythe need for special equipment of pediatric recovery department,and particularly an experienced team of pediatric anesthesiologists.Lately, the negative effect of anesthesia in children is beingincreasingly discussed. It's a question for anesthesiology societieswhose conclusion will affect the future of this method. Currentlywe are awaiting the results of large multicenter studies [25].Recently published works both confirm and refute the negativeeffect of anesthesia. According to Wilder et al. [26], childrenreceiving multiple anesthesia demonstrate increasing incidence of

Fig. 5. 5-year-old boy with UCLP (front view)- GOSLON score 4.

Fig. 6. 5-year-old boy with UCLP (side view)- GOSLON score 4.

Fig. 8. 5-year-old boy with UCLP (occlusion-side view)- GOSLON score 4.

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e77 75

learning disabilities. Conversely, the Czech study by Petr�a�ckov�aet al. [27] observed that no negative impact of earlier anesthesia onintelligence quotient in 3e7 year old children with cleft lip wasrecorded compared to the group of patients on which later anes-thesia was performed.

In our institution we prefer the following treatment protocol:primary cleft lip repair is performed from the second day of life,primary cleft palate repair starting at 6 months of age in patientswith both unilateral and bilateral complete cleft lip and palate. Thisprotocol has been used in our department since 2005, when the

Fig. 7. 5-year-old boy with UCLP (occlusion-front view)- GOSLON score 4.

first cleft lip repair was performed in neonatal period by J. Vokur-kova, MD, in the Czech Republic. On parents' request cleft lip repairmight be postponed to a later age. Early cleft lip surgery enablescleft palate repair soon to be followed in the first year of life. Earlycleft closure has a beneficial effect on infant feeding. The neonatesaccept first nourishment per os mostly in four hours after a lipsurgery. In our department the need for nasogastric tubewas in lessthan 5% cases in 2014 [28].

Plastic surgeons, dealing with cleft palate issues, are used tofinding the delicate balance between the pressure to perform anearlier cleft palate repair because of the right development ofspeech, and a postponed surgery in order not to affect maxilla'sgrowth [29]. Cleft palate repair is essential for creating correctspeech stereotypes and it is difficult to correct once acquiredcompensatory speech patterns [30]. In addition to poor speechdevelopment, feeding problems are observed in cases of late cleftpalate closure. Especially the transition from liquid to solid food cancause many difficulties for patients as well as for parents. Earlypalate surgery enables timely introduction of non-milk-based foodsinto the infant's diet. However, in some studies authors advocatemaxilla's growth over speech development [31]. As a result somesurgeons choose the middle path and prefer two stage palateclosure; at first, closure of soft palate before the twelfth month oflife, and hard palate closure following later on [32].

There are many rating systems for evaluation maxilla's growthand dental arch constriction available [33]. The GOSLON Yardstick[8], the 5 Year Olds' Index [34] or modified Huddart/Bodenhamscoring system [35,36] are most commonly utilized. The GOSLONYardstick was designed by Mars et al. in 1987 and until present day

Table 4GOSLON score results.

Score GOSLON Yardstick

Examiner 1 Examiner 2

1 3 (13.04%) 3 (13.04%)2 11 (47.83%) 12 (52.17%)3 7 (30.43%) 5 (21.74%)4 2 (8.70%) 3 (13.04%)5 0 (0.00%) 0 (0.00%)Mean 2Average 2.35

Kappa value Standard error0.867 0.088

Table 5The Goslon Score and comparison with other cleft center results.

Articleno.

Author, year of publication/[Reference no.]

Country No. ofpatients

Mean age ofpatients (yrs)

Sample GOSLON 1(%)

GOSLON 2(%)

GOSLON 3(%)

GOSLON 4(%)

GOSLON 5(%)

MeanGOSLON

1 Noverraz, 1993/[43] TheNetherlands

77 4.3 22 58 18 1 1 2.2

2 Williams, 2001/[44] UnitedKingdom I

223 5 5 24 34 18 19 3.2

3 Mars, 2006a/[39] UnitedKingdom II

94 5 20 52 27 1 0 2.1

4 Lilja, 2006a/[45] Sweden 94 5 20 57 26 2 0 2.25 Alam, 2008/[46] Japan 140 6.85 3 11 65 15 6 3.16 Wojtaszek-Slominska, 2010/[47] Poland 120 5.3 A 0 18 41 16 25 3.5

B 41 22 25 8 4 2.17 Southall, 2012/[48] Australia 66 6 2 18 45 32 3 3.178 Kajii, 2013/[49] Japan 135 6.9 2 12 64 16 6 3.139 Koshikawa-Matsunob, 2014/[50] Japan 74 7.0 x x x x x 2.9910 Koskova (presented study) Czech

Republic23 5.3 13 50 26 10 0 2.35

a Approximated Goslon scores read from the graph.b Only mean Goslon score reported.

0.00 1.00 2.00 3.00 4.00

Poland_A_2010 (N = 56)

United_Kingdom_2001 (N = 223)

Australia_2012 (N = 66)

Japan_2013 (N = 135)

Japan_2008 (N = 140)

Japan_2014 (N = 74)

Czech_Republic_2016 (N = 23)

The_Netherlands_1993 (N = 77)

Sweden_2006 (N = 94)

United_Kingdom_2006 (N = 94)

Poland_B_2010 (N = 64)

Fig. 9. Comparison of mean GOSLON scores with published reports from other cleftcenters.

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e7776

it is considered to be one of the best available and the most widelyused system for assessing surgical outcomes [37,38]. Initially theGoslon Yardstick was created for evaluating of 10 year-old UCLPpatients' dental models. However, applying this method for ratingin younger children was justified by Mars et al. in 2006 [39]. TheGoslon Yardstick has been used in many studies, including theEurocleft comparative studies of patients with UCLP [15,40]. TheGoslon Yardstick assesses malocclusions in patients with UCLP. Itsreliability is comparable to the 5-year-old index by Attack [39]. The

0% 20% 40

Japan_2008 (N = 140)

Japan_2013 (N = 135)

Poland_A_2010 (N = 56)

Australia_2012 (N = 66)

United_Kingdom_2001 (N = 223)

Poland_B_2010 (N = 64)

Czech_Republic_2016 (N = 23)

United_Kingdom_2006 (N = 94)

Sweden_2006 (N = 94)

The_Netherlands_1993 (N = 77)

Fig. 10. Distribution of GOSLON sc

Goslon reliability by comparison with another frequently usedscoring system, modified Huddart/Bodenham (MHB) [37], is lower.The major advantage of Goslon Yardstick is also its most commonuse in published works in the last twenty years [41] and as a result,there is a great level of comparison with other cleft centers avail-able [15,42]. Our treatment protocol, neonatal cleft lip repair andone-stage palate repair in the first year of life, was found to producethe Goslon score that approximated the results of other cleft cen-ters worldwide. A comparison with other cleft centers is shown inTable 5 and Figs. 9 and 10.

A new testing sample for evaluation of speech outcomes inCzech language was created. In the study most of patients wererated hypernasality of 3 in 5-point scale, which represents anaverage outcome. Articulation (mean value 2.07) and speechintelligibility (mean value 1.93) showed good speech outcomes. Thedisadvantage of our study is a small number of participants. Theevaluation of UCLP patients proceeds and more data will be pre-sented in the future.

5. Conclusion

A new speech testing sample for Czech language was createdand assessed as sufficient for purpose of speech outcome rating.Hypernasality of mean value 2.79, articulation of 2.07 and speechintelligibility 1.93 on the 5-point scale showgood speech outcomes.The mean Goslon score of 2.35 is comparable with other cleft

% 60% 80% 100%

GOSLON_1+2 (%)

GOSLON_3 (%)

GOSLON_4+5 (%)

ores for various cleft centers.

O. Ko�skov�a et al. / International Journal of Pediatric Otorhinolaryngology 87 (2016) 71e77 77

centers. In summary, this study shows that our concept seems to bea proper approach to treating cleft patients based on the resultsachieved in terms of occlusion and speech outcomes of 5-year-oldUCLP patients.

Funding disclosure

None.

Conflict of interest

None.

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