Schisis

Initiatief: NVPC Aantal modules: 51

Techniek lip- en palatumsluiting bij patiënten met een schisis

Uitgangsvraag

Is er een voorkeur voor een chirurgische techniek bij het sluiten van de lip-, kaak- en/of gehemeltespleet de bij kinderen met een schisis?

Aanbeveling

Positioneer tijdens palatumsluiting de palatummusculatuur naar een meer anatomische positie (verbinding in de mediaan en meer naar posterieur geplaatst) voor een beter resultaat op de spraak.

 

Gebruik een (combinatie van) techniek(en) voor palatumsluiting waarin de chirurg het meest ervaren is zodat het risico op complicaties minimaal is.

 

Gebruik een (combinatie van) techniek(en) voor lipsluiting waarin de chirurg het meest ervaren is voor een optimaal resultaat wat betreft functie en esthetiek met een minimaal risico op complicaties.

 

Stimuleer de discussie over de techniek(en) en uitkomstmaten bij de schisis-chirurgen (special interest groups (SIG’s) NVSCA)*.

 

*Streef daarbij naar een landelijke uniforme definitie welke uitkomstmaten voor schisischirurgie worden gemeten en stimuleer een nationale audit waarin uitkomstmaten per protocol worden vergeleken.

Overwegingen

Level of evidence

The level of evidence regarding different techniques of lip and/or palate repair is limited. Overall, the literature is fragmented due to the number of different techniques and the use of different outcomes measures. The problem of a lack of uniform outcomes measures was also addressed in module Timing of closure.

Summarizing it can be stated that hard and soft palate repair with perfect unhampered growth and optimal speech without VPI remains the holy grail for which no perfect solution has been found so far.

 

For the future the choice of surgical technique ideally should take into account the subphenotype of the cleft, relevant genetic information and an estimate of the potential growth of the maxilla for each individual, but this has not reached clinical practise so far.

 

For lip closure, the evidence is uncertain about the effect the different techniques on esthetics and the other outcome measures were not described in the studies.

In addition, the level of evidence is hampered by the fact that the skill and experience of the surgeon plays an important role in all surgery related studies and no doubt also in CLA/P closure techniques. Small – but perhaps important - technical variations between centres and surgeons performing the same operation do exist (and are the driving force behind innovation). As a result, a von Langenbeck closure in the hand of surgeon or centre A might differ from the same operation by surgeon or centre B. Even within centres true calibration between surgeons is not something commonly done. This all makes measuring outcome and comparison of studies in this field quite complex and lowering evidence levels.

 

Advantages and disadvantages of the different techniques

Palatal closure techniques

This module concerns the techniques of lip and/or palate closure. There is a close relation to the module “Timing of lip and/or palate closure”. Unfortunately, literature does not show enough evidence to support certain techniques of palatal closure. However, we do some suggestions and give some tips in selecting a type of surgical technique.

 

The main used techniques of soft palate closure are straight midline closure of the mucosa with or without lateral relaxing incisions versus double opposing z-plasty according to Furlow. Both techniques are performed by experienced surgeons with acceptable results, but every technique has a learning curve. The advantage of the straight-line closure is its relative ease and it preserves the opportunity to do a second correction through the same scars. The advantage of the Furlow technique is transposition of mucosa increasing the length of the soft palate but at the expense of tissue in width. Therefore, the Furlow technique is less favorite for wider palatal clefts with a higher rate of fistula formation (Williams, 2011). Position of the scars makes revision surgery more complicated in the Furlow technique.

 

Attention should be payed to reconstruction of the muscular sling of the levator veli palatini muscle. Sommerlad (2002) describes many details of this muscular repositioning. The Furlow double opposing z-plasty automatically creates the muscular sling. When a straight midline closure technique of the mucosa is used, separate attention for muscle positioning is indispensable.

 

Frequently used techniques to close the hard palate are the vomer flap (nasal layer only closure), mucoperiosteal flaps axially based on the great palatinal artery or anteriorly and posteriorly based leaf-flaps (Von Langenbeck) and buccal transposition flaps. The advantage of the vomer flap technique is that no scar tissue is made on the palatal bone, which may impair outgrowth of the maxilla. However only the relative thin nasal mucosa is closed and therefore more fragile. The mucoperiosteal flaps are thicker and firmer but require an extensive dissection from the bone which might inhibit full growth of the palatal bone. When the mucoperiosteal flaps are used simultaneous closure of the nasal palatal mucosa can be performed, resulting in a more secure double layer closure. The mucoperiosteal flap technique enables simultaneous closure of soft and hard palate. The advantage of the buccal flap technique is adding of tissue and therefore less dissection of the palatal bone. It can be used for nasal and oral layer closure. Disadvantage is scar formation of the buccal mucosa, it is more time consuming and might need additional surgery on the pedicle of the flap.

Lip closure techniques

Many lip closure techniques are described. Millard, Fisher and straight-line closure are favorite. Unfortunately, literature does not show enough evidence to support certain techniques of lip closure. However, we will do some suggestions in selecting a type of surgical technique. The straight-line technique gives good position of the scar in the -line but lengthening is limited. Straight line closure seems more applicable to incomplete clefts of the lip. Fisher and Millard give better lengthening but a more extensive scar due to z-plasties. Many surgeons have their own variations on those basic techniques. In this context it should be mentioned there is no consensus how to rate the esthetic outcome of lip and nose after closure.

 

In conclusion, none of the mentioned surgical techniques seems to surpass the other, but experience of a surgeon with a certain technique is essential for optimal results.

 

Values and preferences of patients and their parents or guardians

Parents can be confused by the variety of surgical techniques and (sometimes strong) opinions on CLA/P treatment by teams or surgeons. They need unambiguous information and advice. Unfortunately, literature does not provide us with enough scientific evidence to support one technique of surgical treatment for CLA/P over the other. Adversely the experience of a surgeon with certain techniques is essential for optimal results. We advise that each team presents clearly what their vision on CLA/P treatment is, elaborated in a straightforward protocol. Each protocol preferably illustrated by instructive photos and comprehensive movies.

 

Costs/Finances

In general, good results should be achieved in as little surgeries as possible in order to keep the burden on the child, the parents as low as possible, and to keep the health care system within acceptable finances. It is important to properly lay the surgical base in the first year of life, and to keep intermediate interventions during growth to a minimum. The final corrections can be made when the patient has grown out, whereby the patient can then assess and decide for himself or herself what he or she needs or what he or she considers to be desirable.

 

Acceptance, feasibility and implementation

There is still a lot of variation in surgical practice between the cleft teams, although a change seems to take place. Special interest groups within the NVSCA try to tackle this point. Within these groups, knowledge is shared, and appointments are made to standardize clinical practise and to measure outcomes in a uniform way as much as possible. In the Netherlands everyone has equal access to the cleft teams, so there will be no variation in accessibility of care. The different techniques are also equally feasible to perform, each surgeon has his own experience and expertise. It is important that the surgeon uses the (combination of different) technique(s) in which he or she is most experienced in order to achieve the best result.

 

Rationale

Uit de literatuur blijkt dat er geen duidelijke voorkeur voor een techniek is, meerdere technieken zouden gebruikt kunnen worden voor lip- en palatumsluiting. Er zijn geen duidelijke voor- en nadelen van de diverse technieken. Er wordt een brede aanbeveling gegeven, mede door het gebrek aan wetenschappelijk bewijs, waarbij de ervaring en deskundigheid van de chirurg in acht wordt genomen.

Onderbouwing

The presence of a cleft lip, alveolus and/or palate (CLA/P) causes an abnormal appearance, the inability to close the lip and/or the inability to separate the nasal cavity from the oral cavity. A cleft palate causes feeding and speech problems. A cleft lip is often associated with abnormalities of the nose affecting appearance and causing functional problems. A cleft in the alveolar arch results in orthodontic and dental abnormalities. The different CLA/P phenotypes cause permanent stigmata. The aim of the multidisciplinary treatment of CLA/P is to efficiently reduce these problems to a minimum.

 

Surgery that closes the lip and/or the palate (soft and/or hard) aims to improve function and apperance, and at the same time should not impair growth and induce as few new problems as possible. Many different techniques have been described over time. It is important to know which technique gives the best results in the short term and the long term. While writing the previous edition of the guideline (2018), it appeared that there was a shortage of prospective and randomized studies.

1. Soft palate

1.1 Outcome measure speech (critical)

Very low

GRADE

The evidence is uncertain about the effect of different techniques of soft palate repair on speech related outcomes like hypernasality and nasal emission.

 

Sources: (Abdel-Azziz, 2011; Ganesh, 2015; Henkel, 2004; Williams, 2011)

 

1.2 Outcome measure hearing (critical), 1.3 Outcome feeding capability (critical) and 1.6 Outcome measure esthetics (important)

-

GRADE

There is no GRADE assessment due to lack of randomized studies.

 

1.4 Outcome measure maxillary and midface growth (important)

Very low

GRADE

The evidence is uncertain about the effect of the vomer flap technique (one-layer closure) compared to the two-flap technique on dental arch relationship (measured at the age of mixed dentition).

 

Sources: (Ganesh, 2015)

 

1.5 Outcome measure postoperative complications (important)

Very low

GRADE

Postoperative complications, in the sense of fistulae, may occur using different techniques, however, the evidence is uncertain about the effects of the techniques on the incidence and type of fistulae.

 

Sources: (Abdel-Azziz, 2011; Ganesh, 2015; Henkel, 2004; Williams, 2011)

 

2. Hard palate

2.1 Outcome measure speech (critical), 2.2 Outcome measure hearing (critical) 2.3 Outcome feeding capability (critical) and 2.6 Outcome measure esthetics (important)

-

GRADE

There is no GRADE assessment due to lack of studies.

 

2.4 Outcome measure maxillary and midface growth (important)

Very low

GRADE

The evidence is uncertain about the effect of one-flap versus the two-flap techniques for closing the hard palate at approximately 3 months of age on dental arch relationships and maxillary arch dimensions after 5 years.

 

Sources: (Rossell-Perry, 2017)

 

2.5 Outcome measure postoperative complications (important)

-

GRADE

There is no GRADE assessment due to events.

 

Sources: (Rossell-Perry, 2017)

 

3. Lip closure

3.1 Outcome measure speech (important), 3.2 Outcome feeding capability (important), 3.3 Outcome measure maxillary and midface growth (important), 3.4 Outcome measure postoperative complications (critical)

-

GRADE

There is no GRADE assessment due to lack of studies.

 

3.5 Outcome measure esthetics (critical)

Very low

GRADE

The evidence is very uncertain about the effect of different techniques of lip repair on esthetics.

 

Sources: (Chowdri, 1990; De Silva Amaratunga, 2004; Williams, 2011)

1. Palatum molle/ soft palate

Description of studies

A total of four randomized controlled trials (RCTs) were included: Abdel – Aziz (2011); Henkel, (2004); Ganesh (2015); Williams (2011).

 

The study of Abdel-Aziz and Ghandour (2011) is a RCT that compares the effects of the Furlow double opposing Z-plasty (n=22) and the Wardill – Kilner V-Y (n=24) pushback technique in terms of velopharyngeal outcome and speech in patients with a cleft of the soft palate and no other congenital anomalies. Age at surgery was 11-36 months. All cases were followed for at least 1 year. Flexible nasopharyngoscopy and perceptual speech resonance evaluation were used to assess the velopharyngeal closure and speech outcome respectively.

 

The study of Henkel (2004) is a RCT in which the effects of soft palate closure using the wave-line technique in the intravelar veloplasty (n=12) are compared to the classic intravelar veloplasty (n=12) in terms of speech outcomes in patients with complete cleft of the soft palate. Patients were randomly assigned to one or the other group following a previously determined succession. Surgery was performed at the age of 10-12 months. Speech was investigated at the age of 4 years by a speech pathologist blinded for the technique.

 

Ganesh (2015) describes a RCT to evaluate the effects of two different surgical protocols (vomer flap and separate soft palate closure versus two-flap technique) on dental arch relationship, speech outcomes and the occurrence of fistula during mixed dentition (7-10 years). Out of 200 patients recruited only 179 patients completed the treatment for lip and palate repair. Out of those 179 patients, 91 patients were allocated to the VF Group and 88 patients to the TF group. However, only 85 patients in total were seen at the follow up during the period of mixed dentition. In the vomer flap group (n=40), the cleft lip was repaired using the Millard technique along with nose correction. After 6 months, soft palate repair was carried out with sharp separation of the muscle fibers from the enveloping oral and the nasal mucosa and from the hard-palatal shelves. The tensor tendon was released just medial to the hamulus, followed by retro positioning and plication of muscle bundles along the midline. In the two-flap group (n=45), cleft lip was repaired by the Millard technique with nose correction, and anterior palate repair up to the incisor foramen. Six months later, the palate was repaired with two-flap palatoplasty. Both in the two-flap group and the vomer flap group the same technique of soft palate closure was followed. Greater part of the velar muscles (Tensor veli palatini & Levator veli palatini) was dissected free from its abnormal attachments followed by recreation of the muscular sling, retro positioning, and plication of muscle bundles along the midline. All operations were performed by the same plastic surgeon.

 

Williams (2011) describees a RCT in which different surgical techniques and different timings of surgery for complete cleft palate were compared in terms of speech outcome and risk of palatal fistulae in patients with a complete unilateral cleft lip and palate. A 2x2x2 factorial clinical trial was used in which each subject was randomly assigned to 1 of 8 groups: 1 of 2 different lip repairs (Spina versus Millard), 1 of 2 different palate repairs (von Langenbeck versus Furlow) and 1 of 2 different ages at time of palatal surgery (9-12 months versus 15-18 months). All surgeries were performed by the same 4 surgeons. A total of 181 patients were operated at 9-12 months (Spina – Furlow = 35, Millard – Furlow = 43, Spina – Langenbeck = 51, Millard – Langenbeck = 52) and 195 at 15-18 months (Spina – Furlow = 48, Millard – Furlow = 47, Spina – Langenbeck = 46, Millard – Langenbeck = 54). Children were followed for at least the age of 4 years.

 

Comparison 2. Palatum durum/ hard palate

Description of study

One study is included in this literature summary that described hard palate surgery (Rossell-Perry, 2017).

 

The article of Rossell-Perry (2017) a prospective, randomized, single-blind controlled trial describes the effect of relaxing incisions on maxillary growth after primary palatoplasty in patients with unilateral cleft lip and palate at the age of five. Cleft lip repair was performed in all patients at approximately 3 months of age using the two-flap (n=72) or one-flap method (n=70) according to their usual surgical protocol: upper rotation advancement plus double unilimb Z-plasty or triple unilimb Z-plasty. The one-flap palatoplasty differed from the two-flap technique using a relaxing incision on the cleft side only. Extension of the subperiosteal dissection was the same in both groups. The soft palate surgical treatment was likewise identical with both techniques. All operations were performed by the same plastic surgeon. Dental arch relationships and maxillary arch dimensions were evaluated at the age of five. The dental arch was rated from 1 (“excellent”) to 5 (“very poor”) and maxillary arch was evaluated as (1) intercanine distance = distance between the canine mesiobuccal cusp tips, (2) intermolar distance = distance between the second molar mesiobuccal cusp tips, and (3) maxillary length = distance in the midline from a point between the incisors to the posterior border of the maxilla.

 

Comparison 3. Lip closure

Description of studies

Three randomized studies comparing different lip closure techniques were identified; (Chowdri, 1990; De Silva Amartunga, 2004, Williams. 2011).

 

Chowdri (1990) is a randomized comparative study performed in India in which rotation advancement lip repair as described by Millard (n=58) is compared to triangular flap lip repair as described by Randall (n=50) in terms of esthetic results and complications. Age at lip repair was 3 years. Patients were followed for 1 to 6 years. Esthetics were evaluated independently by 3 examiners, each scoring surgical results on a 0 to 10 scale for 10 aspects of lip (50 points) and nose (50 points), making a total assessment of 100 points for the 10 components studied.

 

De Silva Amaratunga (2004) describes an RCT in which the esthetic results of unilateral lip repair are compared for Millard’s method (n=18), Cronin’s method (n=21) and a combination of the two methods (n=20). Lip repair was performed at the age of 2 to 6 months. The results of the repair were assessed 3 months after surgery. Esthetic results were assessed using the Cleft Lip Component Symmetry Index (0 to 100, with 100 points indicating perfect symmetry).

 

Williams (2011) describes a RCT which is mentioned in the section on palatal closure above. In this study not only the surgical techniques for closure of palate where compared but also the surgical closure of lips with 2 different techniques. A 2x2x2 factorial clinical trial was used in which each subject was randomly assigned to 1 of 8 groups: 1 of 2 different lip repairs (Spina versus Millard), 1 of 2 different palate repairs (von Langenbeck versus Furlow) and 1 of 2 different ages at time of palatal surgery (9 to 12 months versus 15 to 18 months). All surgeries were performed by the same 4 surgeons. A total of 181 patients were operated at 9 to 12 months (Spina - Furlow = 35, Millard - Furlow = 43, Spina - Langenbeck = 51, Millard - Langenbeck = 52) and 195 at 15 to 18 months (Spina - Furlow = 48, Millard - Furlow = 47, Spina - Langenbeck = 46, Millard - Langenbeck = 54). Children were followed for at least the age of 4 years.

 

Results

The different techniques and the different outcomes in the included studies are presented in Table 1.

 

Table 1 different comparisons and different techniques in the included studies

Study

Techniques

Results

Experimental group

Control group

Experimental group

Control group

1. Soft palate

Abdel-Azziz, 2011

Furlow double opposing Z-plasty (n=22)

Wardill – Kilner V-Y pushback technique (n=24)

Speech

Nasality: 0.88 ± 1.01

 

Nasal emission: 0.92 ± 1.1

 

Complications: Fistulae N=2

Speech

Nasality: 0.27 ± 0.55

 

Nasal emission: 0.36 ± 0.73

 

Complications: Fistulae N=0

Ganesh, 2015

vomer flap (Millard technique for the lip along with nose correction) (n=40)

two-flap technique for the whole palate (Millard technique with nose correction, and anterior palate repair up to the incisor foramen) (n=45)

Speech

Hypernasality

Normal: 11.8%

Mild: 70.6%

Moderate: 17.6%

 

Weak oral pressure

words (present): 41.2%

sentences (present): 41.2%

 

maxillary and midface growth

Dental arch relation: 2.15 (0.662)

Speech

Hypernasality

Normal: 20.5%

Mild: 76.9%

Moderate: 2.6%

 

Weak oral pressure

words (present): 15.4%

sentences (present): 15.4%

 

maxillary and midface growth

Dental arch relation: 2.49 (0.757)

Henkel, 2004

wave-line technique in the intravelar veloplasty (n=12)

classic intravelar veloplasty (n=12)

Speech

Compensatory grimacing when speaking: 1/12

(α-ι) test negative: 12/12

Sounds: /l/, /n/, /d/, /t/ normal: 6/10

Sounds /z/, /s/ normal: 6/10

 

Complications

1 patient with wound dehiscence in the oral mucosa

Speech

Compensatory grimacing when speaking: 8/12

(α-ι) test negative: 8/12

Sounds: /l/, /n/, /d/, /t/ normal: 3/12

Sounds /z/, /s/ normal: 4/12

 

Complications

No complications

Williams, 2011

Spina-Furlow 9-12 months (n=35)

Spina – Langenbeck 9-12 months (n=51)

Spina-Furlow 15-18 months (n=48)

Spina – Langenbeck 15-18 months (n=46)

Millard -Furlow 9-12 months (n=43)

Millard – Langenbeck 9-12 months (n=52)

Millard - Furlow 15-18 months (n=47)

Millard – Langenbeck 15-18 months (n=54)

Speech

Hypernasality (Von Langenbeck versus Furlow)

OR 0.54 (95% CI: 0.31 – 0.95) p=0.014

Nasal air emission (Von Langenbeck versus Furlow)

OR: 0.72 (95% CI 0.45 – 1.15) p=0.12

 

Complications

In total 37/269 (14%) patients operated by von Langenbeck developed fistula, versus 44/190 (23%) in the Furlow operation group. The odds ratio for fistula formation in the von Langenbeck versus the Furlow group was 1.93 (95% CI: 1.12 – 3.14, p=0.008).

 

 

 

 

 

 

 

 

 

 

2. Hard palate

Rossell-Perry, 2017

two-flap upper rotation advancement plus double unilimb Z-plasty or triple unilimb Z-plasty (n=72)

one-flap upper rotation advancement plus double unilimb Z-plasty or triple unilimb Z-plasty and use of a relaxing incision on the cleft side only (n=70)

maxillary and midface growth

5-year-olds’ index: 2.57 ± 1.08

 

maxillary and midface growth

5-year-olds’ index: 2.80 ± 1.91

3. Lip closure

Chowdri, 1990

Millard (n=58)

triangular flap lip repair as described by Randall (n=50)

Esthetics

Lip: 38 ± 5 (possible range 0-50)

Nose: 34 ± 4 (possible range 0-50)

Lip + Nose: 71 ± 10 (possible range 0-100)

Esthetics

Lip: 39 ± 5 (possible range 0-50)

Nose:34 ± 4 (possible range 0-50)

Lip + Nose: 73 ± 12 (possible range 0-100)

De Silva Amartunga, 2004

Millard’s method (n=18)

Cronin’s method (n=21)

combination of the two methods (n=20)

Esthetics

Cupid’s bow height

77

 

Vermillion height:

87

 

 

Nostril height symmetry:

93

 

Nostril width:

96

Esthetics

 

Cupid’s bow height

Cronin 86

Combined: 87

Vermillion height:

Cronin: 97

Combined: 97

 

Nostril height symmetry:

Cronin: 93

Combined: 92

Nostril width:

Cronin: 99

Combined: 95

Williams, 2011

See above

 

 

 

 

 

1. Palatum molle/ soft palate

1.1 Outcome measure speech (critical)

Four studies (Abdel-Azziz and Ghandour (2011); Ganesh (2015); Henkel (2004); Williams (2011)) assessed speech. Due to heterogeneity of the different techniques evaluated in the studies and the different outcome measures, it was not possible to pool the data.

 

Abdel-Azziz and Ghandour (2011) described in 46 patients that velopharyngeal closure and speech outcome were statistically better after Furlow Z-plasty than after the V-Y pushback procedure. Auditory perceptual assessment for nasality was 0.88 ± 1.01 in the Furlow group and 0.27 ± 0.55 in the V-Y pushback group (p=0.035). Nasal emission was 0.92 ± 1.1 in the Furlow group versus 0.36 ± 0.73 in the V-Y pushback group (p=0.049). Glottal articulation was 1.13 ± 1.04 in the Furlow group and 0.50 ± 0.74 in the V-Y pushback group (p=0.029). There was no statistically significant difference in pharyngealization of fricatives and speech intelligibility between the groups.

 

Ganesh (2015) reported speech outcomes of patients receiving vomer flap (VF) technique or two-flap technique (TF). In the VF group of 91 patients 36 needed releasing incisions. It was observed that the researchers were able to achieve less posteriorization of the velum in VF group when compared to the TF group (personal comment author upon e-mail request from working group May 2020). The results of the speech outcomes were obtained from only 34 patients in the VF group and 39 in the TF group, for various reasons. The results showed that 11.8% of patients in the vomer-flap group had normal resonance and 17.6% had moderate hypernasality. In the two-flap group 20.5% of patients had normal resonance and 2.6% showed moderate hypernasality, resulting in a RR of 6.88 (95% CI 0.87 to 54.35) for moderate hypernasality. Weak oral pressure consonants were perceived in 41% and 15% of the vomer flap and two-flap group, respectively (RR 2.68 (95% CI 1.16 to 6.19)).

 

Henkel (2004) showed in 24 patients that compensatory grimacing when speaking was observed in 1/12 patients in the wave-line veloplasty group versus 8/12 in the classic intravelar veloplasty group (p<0.05). A sound difference was observed in speech with a closed and open nose in 12/12 of the wave-line veloplasty group and 8/12 in the classic intravelar veloplasty group (p<0.05). Articulation of alveolar sounds was judged normal in significantly more subjects in the wave-line veloplasty group (6/10 (2 children too playful for examination)) versus the classic intravelar veloplasty group (3/12, p<0.05). They concluded that the waveline technique seems to be superior, however group size is small.

 

Williams (2011) reports in 376 subjects that patients operated using the Von Langenbeck technique had an 0.54 OR (95% CI 0.31 to 0.95, p=0.014) for hypernasality and a 0.72 OR (95% CI: 0.45 to 1.15, p=0.12) for nasal air emission when compared to the Furlow technique.

 

Level of evidence of the literature

The level of evidence regarding the outcome measure speech started at high as it was based on randomized controlled trials. The level of evidence was downgraded by three levels to very low due to the risk of bias (-1, concealment allocation) and limited number of included patients (-2, imprecision).

 

1.2 Outcome measure hearing (critical)

The outcome hearing was not reported in the included studies.

 

Level of evidence of the literature

The level of evidence regarding the outcome measure hearing was not assessed due to lack of studies.

 

1.3 Outcome feeding capability (critical)

The outcome feeding capacity was not reported in the included studies.

 

Level of evidence of the literature

The level of evidence regarding the outcome measure feeding capacity was not assessed due to lack of studies.

 

1.4 Outcome measure maxillary and midface growth (important)

The dental arch relationship was measured by Ganesh (2015) with GOSLON scores; 1: good growth - 5: very poor growth. GOSLON scores of the 40 patients in the vomer flap group and 45 patients in the two-flap group were based on the intraoral digital photographs examined by a surgeon and an orthodontist, who were not members of the cleft team. These photographs were taken during regular follow-up through the period of mixed dentition (age range 7 to 9 years old). The mean GOSLON score in the vomer flap group was lower than the GOSLON score in the two-flap group (mean difference = -0.34 (95%CI -0.64 to -0.04). In the vomer flap group, 70% of the patients demonstrated good growth (GOSLON scores 1 and 2) and 30% revealed adequate growth (GOSLON score 3). In the TF group, 54% of the patients displayed good growth, 37.7% had the adequate growth category, and 8.8% had poor growth (GOSLON score 4). None of the patients from either group were found to have very poor GOSLON scores (GOSLON score 5).

 

Level of evidence of the literature

The level of evidence regarding the outcome maxillary and midface growth started at high as it was based on a randomized controlled trial. The level of evidence was downgraded by three levels to low due to loss to follow up (-1, risk of bias), and the small study population and crossing the borders of clinical relevance (-2, imprecision).

 

1.5 Outcome measure postoperative complications (important)

Four studies (Abdel-Azziz and Ghandour (2011); Ganesh (2015); Henkel (2004); Williams (2011)) assessed postoperative complications. Due to heterogeneity of the different techniques evaluated in the studies, it was not possible to pool the data.

 

Abdel-Azziz (2011) reports that in total 0/22 fistulas were found in the Furlow group versus 2/24 in the V-Y pushback procedure group (no p-value reported).

 

Ganesh (2015) reported that palatal fistula appeared only in one patient in the vomer flap group (n=40) and did not appear in the two-flap group (n=45).

 

Henkel (2004) reports that 1 patient in the wave-line group developed wound dehiscence in the oral mucosa that healed secondarily without complications (p-value not reported). All other patients healed without complications.

 

In Williams (2011) only fistula rates were compared and were found not to be different between the Spina and Millard repair technique. In total 37/269 (14%) patients operated by von Langenbeck developed fistula, versus 44/190 (23%) in the Furlow operation group. The odds ratio for fistula formation in the von Langenbeck versus the Furlow group was 1.98 (95% CI: 1.16 to 3.07, p=0.010).

 

Level of evidence of the literature

The level of evidence regarding the outcome postoperative complications started at high as it was based on randomized controlled trials. The level of evidence was downgraded by three levels to very low due to the risk of bias (-1, concealment allocation) and limited number of included patients (-2, imprecision).

 

1.6 Outcome measure esthetics (important)

The outcome esthetics was not reported in the included studies.

 

Level of evidence of the literature

The level of evidence regarding the outcome measure esthetics was not assessed due to lack of studies.

 

2. Palatum durum / hard palate

2.1 Outcome measure speech (critical), 2.2 Outcome measure hearing (critical), 2.3 Outcome feeding capability (critical), and 2.6 Outcome measure esthetics (important)

These outcome measures were not reported in the included studies.

 

Level of evidence of the literature

The level of evidence regarding the outcome measures speech, hearing, feeding capability, and esthetics were not assessed due to lack of studies.

 

2.4 Outcome measure maxillary and midface growth (important)

Rossell-Perry (2017) reported dental arch relationships and maxillary arch dimensions after 5 years. Dental arch relationships were assessed using the 5-year-olds’ index ranging from 1 (excellent dental arch relationship) to 5 (very poor dental arch relationship). The mean score for the 5-year-olds’ index was 2.57±1.09 in the two-flap technique group (n=72) and 2.80±1.91 in the one-flap technique group (n=70); these scores were not significantly different between the groups. None of the patients developed a very poor dental arch relationship.

 

Maxillary arch dimensions were measured on the maxillary arch casts (1) intercanine distance = distance between the canine mesiobuccal cusp tips, (2) intermolar distance = distance between the second molar mesiobuccal cusp tips, and (3) maxillary length = distance in the midline from a point between the incisors to the posterior border of the maxilla. The intercanine distance was 27.64±1.57, the intermolar distance was 35.32±1.32 and the maxillary length was 29.63±2.14 in the two-flap group. In the one-flap group the intercanine distance was 27.32±1.87, the intermolar distance was 35.92±1.21 and the maxillary length was 30.02±2.04. These difference between the groups were not significant.

 

Level of evidence of the literature

The level of evidence regarding the outcome maxillary and midface growth started at high as it was based on a randomized controlled trial. The level of evidence was downgraded by two levels due to risk of bias (-1, incomplete accounting of patients loss to follow up) and due to the small study population (-1, imprecision).

 

2.5 Outcome measure postoperative complications (important)

Rossell-Perry (2017) reported an equal number of postoperative palatal fistulas in both the two-flap and one-flap group (n = 2; 2.85%). All fistulas were asymptomatic and located in the middle third of the palate.

 

Level of evidence of the literature

The level of evidence regarding the outcome postoperative complications was not graded due to the small number of events.

 

3. Lip closure

3.1 Outcome measure speech (important), 3.2 Outcome feeding capability (important), 3.3 Outcome measure maxillary and midface growth (important), 3.4 Outcome measure postoperative complications (critical)

These outcome measures were not reported in the included studies.

 

Level of evidence of the literature

The level of evidence regarding the outcome measures speech, hearing, and feeding capability, maxillary and midface growth, and postoperative complications were not assessed due to lack of studies.

 

3.5 Outcome measure esthetics (critical)

Three studies (Chowdri (1990); De Silva Amaratunga (2004); Williams (2011)) assessed esthetics. Due to heterogeneity of the different techniques evaluated in the studies, it was not possible to pool the data.

 

Chowdri (1990) report that the esthetic scores were similar in the patients treated with rotation advancement repair (71 ± 10) and triangular flap repair (73 ± 12, p> 0.50). There was also no significant difference for the lip-scores alone (p>0.10) or the nose-scores alone (p>0.80) between the two surgical techniques. Regarding postoperative complications, this study reports that 5/58 (9%) of the patients in the rotation flap advancement group developed scar hypertrophy compared to 2/50 (4%) in the triangular flap group (p>0.10). Furthermore, 4 patients in the rotation advancement group developed wound dehiscence, compared to 0 patients in the triangular flap group (p-value not reported).

 

De Silva Amaratunga (2004) reports that the Cleft Lip Component Symmetry Index score of philtral height, vermillion height, and Cupid's bow height achieved with the combined method was comparable to that achieved with Cronin's method and was superior to that obtained with Millard’s method (p< 0.01). Further, the combined method achieved a Cleft Lip Component Symmetry Index score for the philtral width that was not significantly different from that of Millard's method and better than that of Cronin’s method (p< 0.01).

 

In Williams (2011) only fistula rates were compared and were found not to be different between the Spina and Millard repair techniques. The esthetic result of the different lip-closure techniques was not compared in this study.

 

Level of evidence of the literature

The level of evidence regarding the outcome esthetics started at high as it was based on randomized controlled trials. The level of evidence was downgraded by three levels due to due to risk of bias (-1, incomplete accounting of patients loss to follow up) and limited number of included patients (-2, imprecision).

A systematic review of the literature was performed to answer the following question:

What is the effect of different techniques of repairing cleft lip, alveolus and/or palate on speech, hearing, feeding capability, maxillary and midface growth, postoperative complications and esthetics (patient, parent and/or doctor satisfaction)?

 

P: patients with cleft lip, alveolus and/or palate

I: surgical intervention with a specific technique for repairing cleft lip, alveolus and/or palate

C: surgical intervention with another surgical technique for repairing cleft lip, alveolus and/or palate

O: speech, hearing, feeding capability, maxillary and midface growth, postoperative complications, esthetics (patient, parent and/or doctor satisfaction).

 

Relevant outcome measures

For cleft palate repair, the working group considered the following outcome measures as critical for decision making: speech, hearing and feeding capability. Maxillary and midface growth, postoperative complications, and esthetics were considered as important.

 

For cleft lip repair, the working group considered the following outcome measures as critical for decision making: postoperative complications, and esthetics were selected as critical.

Speech, feeding capability, and maxillary and midface growth, were considered as important.

 

Studies should report at least one of the outcomes of interest: esthetics (patient, parent and/or doctor satisfaction), speech, feeding capability, postoperative complications. For the outcome speech, a follow-up until at least the age of four years. A follow-up length of at least one year was deemed sufficient for the outcome hearing. When papers reported a shorter follow-up time they were excluded.

 

A priori, the working group did not define the outcome measures listed above but used the definitions used in the studies.

 

The working group defined the criteria for minimal clinically (patient) important difference for the dichotomous outcome measures; RR < 0.80 of > 1.25)

 

No a priori criteria were set for the continuous outcome measures because it largely depends on its context. If no information was available about the clinically important difference of the outcome measure, a difference of ten percent between the groups was defined as a minimally clinically important difference.

 

Search and select (Methods)

A systematic search had been performed for the 2018 edition of the guideline in the databases Medline (through OVID), Embase and the Cochrane Library on December 3rd, 2014. This search identified 519 references, and 39 references were selected after reviewing title and abstracts. After checking eligibility of full text articles, 25 studies were excluded and 14 studies were included in the summary of literature, of which seven RCTs, and seven observational studies. To update the previous search, the databases Medline (via OVID) and Embase (via Embase.com) were searched with previous search terms until January 1st, 2020. The detailed search strategy is depicted under the tab Methods. The systematic literature search resulted in 529 new hits. Studies were selected based on the following criteria: studies including patients with CLA/P were selected if they compared two different operative techniques in terms of closure of the soft palate. A total 14 studies (8 reviews and 6 RCTs) were initially selected based on title and abstract screening. After reading the full text, the 8 review studies and 4 RCTs were excluded (see the table with reasons for exclusion under the tab Methods), and 2 RCTs were included and added to the six RCTs of the previous search who met the selection criteria above.

 

Results

Two studies were added to the 2018 version of the analysis of the literature describing (lip and) palate closure; one study described the closure of palatum molle/soft palate and one study described the closure of palatum durum/hard palate. No studies describing lip closure were added. To distinguish between lip and palate closure and between hard and soft palate, the literature analyses is divided in three parts:

  1. Palatum molle / soft palate
  2. Palatum durum / hard palate
  3. Lip closure

Important study characteristics and results are summarized in the evidence tables. The assessment of the risk of bias is summarized in the risk of bias tables.

  1. Abdel-Aziz, M., & Ghandour, H. (2011). Comparative study between VY pushback technique and Furlow technique in cleft soft palate repair. European Journal of Plastic Surgery, 34(1), 27-32.
  2. Chowdri, Nisar Ahmad, Mohd Ashraf Darzi, and Mufti M. Ashraf. "A comparative study of surgical results with rotation-advancement and triangular flap techniques in unilateral cleft lip." British journal of plastic surgery 43.5 (1990): 551-556.
  3. de Silva Amaratunga, Nihal Asoka. "Combining Millard's and Cronin's Methods of Unilateral Cleft Lip Repair - a Comparative Study." Asian Journal of Oral and Maxillofacial Surgery 16.1 (2004): 5-9.
  4. Ganesh P, Murthy J, Ulaghanathan N, Savitha VH. (2015). A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition. J Craniomaxillofac Surg;43(6):790-5.
  5. Henkel KO, Dieckmann A, Dieckmann O, Lenz JH, Gundlach KK. (2004). Veloplasty using the wave-line technique versus classic intravelar veloplasty. Cleft Palate Craniofac J;41(1):1-4.
  6. Rossell-Perry P, Cotrina-Rabanal O, Figallo-Hudtwalcker O, Gonzalez-Vereau A. (2017). Effect of Relaxing Incisions on the Maxillary Growth after Primary Unilateral Cleft Palate Repair in Mild and Moderate Cases: A Randomized Clinical Trial. Plast Reconstr Surg Glob Open.16;5(1):e1201.
  7. Sommerlad BC, Mehendale FV, Birch MJ, Sell D, Hattee C, Harland K. (2002). Palate Re-Repair Revisited. The Cleft Palate-Craniofacial Journal;39(3):295-307.
  8. Williams, W. N., Seagle, M. B., Pegoraro-Krook, M. I., Souza, T. V., Garla, L., Silva, M. L.,... & Whitaker, M. E. (2011). Prospective clinical trial comparing outcome measures between Furlow and von Langenbeck palatoplasties for UCLP. Annals of plastic surgery, 66(2), 154-163.

Research question: What is the effect of different techniques of repairing cleft lip, alveolus and/or palate on speech, hearing, diet, postoperative esthetical complications (patient, parent and/or doctor satisfaction)?

Study reference

Study characteristics

Patient characteristics 2

Intervention (I)

Comparison / control (C) 3

 

Follow-up

Outcome measures and effect size 4

Comments

Palatum molle

Abdel-Aziz, 2011

Type of study: randomized controlled trial

 

Setting: outpatients

 

Country: Egypt

 

Source of funding: not reported

Inclusion criteria:

1) patients with cleft soft palate without any other congenital anomalities

2) treated at participating hospital

 

Exclusion criteria:

-

 

N total at baseline:

Intervention: 24

Control: 22

 

Important prognostic factors2:

For example

age ± SD:

total group: 1 year 4 months (range 11 months – 3 years)

 

Sex:

Total group: 45% M

 

Groups comparable at baseline? unclear

 

Describe intervention (treatment/procedure/test):

 

 

V-Y pushback technique for cleft palate repair

 

Describe control (treatment/procedure/test):

 

 

Furlow double-oppsing Z-plasty for cleft repair

Length of follow-up:

At least 1 year

Until the age of 4 years

 

Loss-to-follow-up:

60 patients included in total (number per group not mentioned)

Speech assessment performed in I: 24, C: 22

Reasons for this not reported

 

Incomplete outcome data:

See above

 

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Means and standard deviations of the auditory perceptual assessment in both treatment groups

 

Nasality

I: 0.88 ± 1.01

C: 0.27 ± 0.55

P=0.035

 

Glottal Articulation

I: 1.13 ± 1.04

C: 0.5 ± 0.74

P=0.029

 

Pharyngealization of fricatives:

I: 1.00 ± 1.18

C: 0.55 ± 1.1

P=0.10

 

Nasal emission:

I: 0.92 ± 1.1

C: 0.36 ± 0.73

P=0.049

 

Speech intelligibility

I: 1.08 ± 1.25

C: 0.59 ± 1.1

P=0.14

 

Fistulae:

I: n=2

C: n=0

p-value not reported

Author’s conclusion: The Furlow Z-plkasty is better than the V-Y pushback technique in repair of clefts involving the soft palate as it has a higher success rate regarding speech outcome and velopharyngeal closure; also it has a lower operative tima and blood loss.

Ganesh, 2015

Type of study: randomized controlled trial

 

Setting: not reported

 

Country: India

 

Source of funding: not reported

Inclusion criteria:

patients with nonsyndromic unilateral CLA/P

 

Exclusion criteria:

children operated on by more than one surgeon were excluded

 

N total at baseline: 200

Intervention: 100

Control: 100

 

Important prognostic factors2:

Mean age lip repair

I: 5.2 mo

C: 6.3 mo

 

Mean age palate repair

I: 12.3 mo

C: 12.9 mo

 

Mean age follow up

I: 7.8 y

C: 8.1 y

 

Sex:

I: 60% M

C: 51% M

 

Groups comparable at baseline? Yes

Describe intervention (treatment/procedure/test):

 

Protocol 1 consisted of the vomer flap (VF), whereby

patients underwent primary lip nose repair and vomer flap for hard palate single-layer closure, followed

by soft palate repair 6 months later

Describe control (treatment/procedure/test):

 

Protocol 2 consisted of the two-flap technique (TF), whereby the cleft palate (CP) was repaired by two-flap technique after primary lip and nose repair

Length of follow-up:

7-10 years

 

Loss-to-follow-up:

115

 

Of the 200 randomized patients, 179 completed the protocol. However, only 85 patients presented for follow-up (I: 40 and C: 45)

 

Incomplete outcome data:

For various reasons, speech samples were obtained from only 34 patients in the VF group and 39 in the TF group.

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Dental arch relation (mean GOSLON Yardstick)

I: 2.15 ± 0.662

C: 2.49 ± 0.757

P=0.032

 

Speech outcomes

Hypernasality

I:

Normal: 11.8%

Mild: 70.6%

Moderate: 17.6%

C:

Normal: 20.5%

Mild: 76.9%

Moderate: 2.6%

P=0.05

 

Weak oral pressure words (present)

I: 41.2%

C: 15.4%

P=0.014

 

Weak oral pressure sentences (present)

I: 41.2%

C: 15.4%

P=0.014

 

Author’s conclusion:

Our results showed marginally better growth outcome in the VF group compared to the TF group. However, the speech outcomes were better in the TF group.

Henkel, 2003

Type of study: randomized trial

 

Setting: outpatients

 

Country: Germany

 

Source of funding: not reported

Inclusion criteria:

1) patients with complete cleft of the soft palate

 

Exclusion criteria:

-

 

N total at baseline:

Intervention: 12

Control: 12

 

Important prognostic factors2:

For example

age ± SD: NR

 

Sex:

NR

 

Groups comparable at baseline? Unclear

 

Describe intervention (treatment/procedure/test):

 

Wave-line technique for intravelar veloplasty

Age: 12 months

 

Labioplasty: 6 months

Repair of hard palate: 4-5 years

Primary bone grafting: 11-13 years

 

Describe control (treatment/procedure/test):

 

Classic intravelar veloplasty

Age: 12 months

 

Labioplasty: 6 months

Repair of hard palate: 4-5 years

Primary bone grafting: 11-13 years

 

Length of follow-up:

Until the age of 4 years

 

Loss-to-follow-up:

No loss to follow-up reported

 

Incomplete outcome data:

In wave-line technique group 2/12 (17%) were too playful for examination on sound tests

 

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Mouth breathing:

I: 4/12

C: 8/12

p-value not reported

 

Compensatory grimacing when speaking

I: 1/12

C: 8/12

P<0.05

 

(α-ι) test negative

I: 12/12

C: 8/12

P<0.05

 

Sounds: /l/, /n/, /d/, /t/ normal

I: 6/10

C: 3/12

P<0.05

 

Sounds /z/, /s/ normal

I: 6/10

C: 4/12

P<0.05

Author’s conclusion:

Primary repair of clefts of the soft palate using the wave-line is straightforward, safe and easy. On the basis of the present results, this technique seems superior to the classic intravela veloplasty.

Rossell-Perry, 2017

Type of study: prospective, randomized, double-blind controlled trial study

 

Setting: Surgical Center Program

Lima

 

Country: Peru

 

Source of funding: not reported

Inclusion criteria:

Peruvian infants with nonsyndromic

complete unilateral cleft lip and palate who were otherwise healthy. Only children with mild or moderate

unilateral cleft lip and palate were included in this study because severe cases require a relaxing incision on the cleft side.

 

Exclusion criteria:

Patients with severe unilateral cleft lip and palates were excluded.

 

N total at baseline:

I: 78

C: 78

 

Important prognostic factors2:

Sex (male):

I: 54%

C: 61%

 

Cleft severity (moderate)’

I: 72%

C: 67%

 

Groups comparable at baseline? Yes

Describe intervention (treatment/procedure/test):

 

The two-flap palatoplasty group underwent Bardach’s

two-flap palatoplasty plus the Sommerlad type of intravelar veloplasty and unilateral uvuloplasty.

 

31% upper rotation advancement + double unilimb Z-plasty

69% triple unilimb Z-plasty

 

All operations were

performed by the same plastic surgeon

 

Describe control (treatment/procedure/test):

 

One flap technique. The one-flap palatoplasty differed from the two-flap

technique by the use of a relaxing incision on the cleft

side only.

 

33% upper rotation advancement + double unilimb Z-plasty

67% triple unilimb Z-plasty

 

All operations were

performed by the same plastic surgeon

Length of follow-up:

5 years

 

Loss-to-follow-up:

I: 6

C: 8

 

Incomplete outcome data:

-

Outcome measures and effect size (include 95%CI and p-value if available):

 

evaluation of maxillary arch dimensions and dental arch relationships using the 5-year-olds’ index, rating from 1 (“excellent”) to 5 (“very poor”).

I: 2.57 ± 1.08

C: 2.80 ± 1.91

P=0.71

Author’s conclusion:

The results arising from this clinical trial do not provide statistical evidence that one technique let us obtain better maxillary development than the other at 5 years. The use of relaxing incisions was not associated with maxillary growth impairment. A technique with limited relaxing incisions does not has better maxillary growth.

Williams, 2009

Type of study: randomized controlled trial

 

Setting: outpatients

 

Country: United States of America / Brazil

 

Source of funding: non-commercial

Inclusion criteria:

1) patients with complete unilateral cleft lip and palate

 

Exclusion criteria:

1) failure of family to return to the hospital at assigned operation date

2) conditions impairing speech development (hearing problems, mental retardation)

 

N total at baseline:

Intervention: 134

Control: 201

 

Important prognostic factors2:

Furlow:

Spina: 83, Millard: 90

Operation at 9-12 months: 78

Operation at 15-18 months: 95

 

Von Langenbeck

Spina: 97, Millard: 106

Operation at 9-12 months: 103

Operation at 15-18 months:100

 

Groups comparable at baseline? Unclear

Describe intervention (treatment/procedure/test):

 

Furlow

 

Describe control (treatment/procedure/test):

 

 Von Langenbeck

Length of follow-up:

Until the age of 4 years

 

Loss-to-follow-up:

Not reported

 

Incomplete outcome data:

Not reported

 

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Hypernasality:

Odds ratio (OR) Von Langenbeck versus Furlow

0.54 (95% CI: 0.31 – 0.95) p=0.014

 

 

Nasal air emission

Odds ratio (OR) Von Langenbeck versus Furlow

0.72 (95% CI 0.45 – 1.15) p=0.12

Author’s conclusion:

In this study the Furlow double opposing Z-palatoplasty resulted in significantly better velopharyngeal function for speech than the von Langenbeck procedure as determined by the perceptual cul-de-sac test of hypernasality.

Lip adhesion

Chowdri, 1990

Type of study: randomized trial

 

Setting: outpatients

 

Country: India

 

Source of funding: not reported

Inclusion criteria:

1) patients with unilateral cleft lip

 

Exclusion criteria:

-

 

N total at baseline:

Intervention: 58

Control: 50

 

Important prognostic factors2:

For example

ag:

I: 3.1

C: 2.9

 

Sex: NR

 

Complete cleft:

I: 38/58 (73%)

C: 30/50 (60%)

 

Associated cleft palate:

I: 39/58 (67%)

C: 32/50 (64%)

 

Groups comparable at baseline? yes

Describe intervention (treatment/procedure/test):

 

 

Rotation advancement

 

Describe control (treatment/procedure/test):

 

 

Triangular flap repair

Length of follow-up:

6 years

 

Loss-to-follow-up:

Follow-up period (years) and number of patients per period (reasons for dropout not described:

1-2: 11

2-3: 22

3-4: 35

4-5: 29

5-6: 11

 

 

Incomplete outcome data:

As above

 

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Comparison scores (esthetic results)

 

Lip

I: 38 ± 5

C: 39 ± 5

p>0.10

 

Nose

I: 34 ± 4

C: 34 ± 4

p>0.80

 

Lip + Nose

I: 71 ± 10

C: 73 ± 12

p>0.50

Author’s conclusion:

No significant difference was found in overall postoperative appearance of lip and nose between rotation-advancement and triangular flap repair. As a result we recommend either technique for unilateral cleft lip repair.

De Silva Armatunga, 2004

Type of study: randomized trial

 

Setting: outpatients

 

Country: Sri Lanka

 

Source of funding: not reported

Inclusion criteria:

1) patients with unilateral cleft lip

 

Exclusion criteria:

-

 

N total at baseline:

Millard: 18

Cronin: 21

Combined: 20

 

Important prognostic factors2:

For example

age ± SD:

unclear (age distribution reported)

 

Sex:

Millard: 61% M

Cronin: 62% M

Combined: 65% M

 

Groups comparable at baseline? Unclear

Describe intervention (treatment/procedure/test):

 

Millard procedure

 

 

 

Cronin procedure

 

 

Describe control (treatment/procedure/test):

 

Method combining Millard an Cronin Procedure

Length of follow-up:

3 months

 

Loss-to-follow-up:

Not reported

 

Incomplete outcome data:

Not reported

 

 

Outcome measures and effect size (include 95%CI and p-value if available):

 

Cleft Lip Component Symmetry Index score

 

Cupid’s bow height

Millard: 77

Cronin 86

Combined: 87

P<0.01 for Millard versus other 2 groups

 

Vermillion height:

Millard: 87

Cronin: 97

Combined: 97

P<0.01 for Millard versus other 2 groups

 

Nostril height symmetry:

Millard: 93

Cronin: 93

Combined: 92

P>0.05, reduced in all groups

 

Nostril width:

Millard: 96

Cronin: 99

Combined: 95

p>0.05 for Cronin versus other 2 groups

Author’s conclusion:

The advantages of Millard’s and Cronin’s methods, which are the most widely used methods of cleft lip repair; could be retained and the disadvantages eliminated to a degree by combining the 2 methods. A basic muscle repair could also be included in the new combined method.

Williams, 2009

See above

 

 

 

 

 

 

CLA/P: cleft lip, alveolus and/or palate

Notes:

  1. Prognostic balance between treatment groups is usually guaranteed in randomized studies, but non-randomized (observational) studies require matching of patients between treatment groups (case-control studies) or multivariate adjustment for prognostic factors (confounders) (cohort studies); the evidence table should contain sufficient details on these procedures.
  2. Provide data per treatment group on the most important prognostic factors ((potential) confounders).
  3. For case-control studies, provide sufficient detail on the procedure used to match cases and controls.
  4. For cohort studies, provide sufficient detail on the (multivariate) analyses used to adjust for (potential) confounders.

Risk of bias table for intervention studies (randomized controlled trials)

Research question: What is the effect of different techniques of repairing cleft lip, alveolus and/or palate on speech, hearing, diet, postoperative esthetical complications (patient, parent and/or doctor satisfaction)?

Study reference

 

 

 

 

(first author, publication year)

Describe method of randomisation1

Bias due to inadequate concealment of allocation?2

 

 

 

 

(unlikely/likely/unclear)

Bias due to inadequate blinding of participants to treatment allocation?3

 

 

(unlikely/likely/unclear)

Bias due to inadequate blinding of care providers to treatment allocation?3

 

 

(unlikely/likely/unclear)

Bias due to inadequate blinding of outcome assessors to treatment allocation?3

 

(unlikely/likely/unclear)

Bias due to selective outcome reporting on basis of the results?4

 

 

(unlikely/likely/unclear)

Bias due to loss to follow-up?5

 

 

 

 

 

 

(unlikely/likely/unclear)

Bias due to violation of

intention to treat analysis?6

 

 

 

 

(unlikely/likely/unclear)

 

Palatum molle

Abdel-Aziz, 2011

“randomly classified into two equal groups

Likely

Likely

Likely

Likely

Unlikely

Likely

Unlikely

 

Ganesh, 2015

Randomization was

done by allocation concealment, whereby 200 chits were put in a

box (100 for each group) and the parent or guardian was asked to pick one chit 1 day before the surgery.

Unlikely

Unlikely

Unlikely

Unlikely

Unlikely

Unclear, it is unclear what the consequences are of the large group that was lost to follow-up.

Unclear

 

Henkel, 2003

Each patient was assigned to one of the two groups following a previously determined succession

Likely

Likely

Unlikely

Unlikely

Unlikely

Unlikely

Unlikely

 

Williams, 2009

Block randomization plan for 4 surgeons: each sequential set assigned to a surgeon was assigned to the 8 study groups at random

Likely

Likely

Unlikely

Unlikely

Unlikely

Unclear

Unlikely

 

Palatum durum

Rossell-Perry, 2017

Blockrandomization: Sequence generation

for the randomized group allocation was accomplished

through the use of computer-generated random numbers.

A block size of 6 and allocation ratio of 2:1 were used. The

group assignment was protected in a sealed envelope,

which was opened by the surgeon just before surgery.

Unlikely

Unlikely

Unlikely

Unlikely

Unlikely

Unclear

Unclear

 

Lip adhesion

Chowdri, 1990

“simple random sampling procedure”

Likely

Likely

Unlikely

Likely

Unlikely

Unclear

Unclear

 

De Silva Armatunga, 2004

“randomly allocated”

Likely

Likely

Unlikely

Likely

Unlikely

Unclear

Unclear

 

Williams, 2009

See above

 

 

 

 

 

 

 

 

  1. Randomisation: generation of allocation sequences have to be unpredictable, for example computer generated random-numbers or drawing lots or envelopes. Examples of inadequate procedures are generation of allocation sequences by alternation, according to case record number, date of birth or date of admission.
  2. Allocation concealment: refers to the protection (blinding) of the randomisation process. Concealment of allocation sequences is adequate if patients and enrolling investigators cannot foresee assignment, for example central randomisation (performed at a site remote from trial location) or sequentially numbered, sealed, opaque envelopes. Inadequate procedures are all procedures based on inadequate randomisation procedures or open allocation schedules.
  3. Blinding: neither the patient nor the care provider (attending physician) knows which patient is getting the special treatment. Blinding is sometimes impossible, for example when comparing surgical with non-surgical treatments. The outcome assessor records the study results. Blinding of those assessing outcomes prevents that the knowledge of patient assignement influences the proces of outcome assessment (detection or information bias). If a study has hard (objective) outcome measures, like death, blinding of outcome assessment is not necessary. If a study has “soft” (subjective) outcome measures, like the assessment of an X-ray, blinding of outcome assessment is necessary.
  4. Results of all predefined outcome measures should be reported; if the protocol is available, then outcomes in the protocol and published report can be compared; if not, then outcomes listed in the methods section of an article can be compared with those whose results are reported.
  5. If the percentage of patients lost to follow-up is large, or differs between treatment groups, or the reasons for loss to follow-up differ between treatment groups, bias is likely. If the number of patients lost to follow-up, or the reasons why, are not reported, the risk of bias is unclear.
  6. Participants included in the analysis are exactly those who were randomized into the trial. If the numbers randomized into each intervention group are not clearly reported, the risk of bias is unclear; an ITT analysis implies that (a) participants are kept in the intervention groups to which they were randomized, regardless of the intervention they actually received, (b) outcome data are measured on all participants, and (c) all randomized participants are included in the analysis.

 

Table of excluded studies

Author and year

Reason for exclusion

Adetayo, 2018

RCT does not meet selection criteria (follow-up too short)

Adetayo, 2019

RCT does not meet selection criteria (follow-up too short)

Bartzela, 2011

Does not meet selection criteria (does not answer research question, compares center protocols, not surgical techniques).

Bichara, 2015

Review does not meet selection criteria (does not answer research question, compares lip surgery versus lip and palate surgery)

Carroll, 2013

Excluded on study design (observational study)

Deshmukh, 2018

RCT does not meet selection criteria (follow-up too short)

Enemark, 1993

Does not meet selection criteria (palatum durum)

Flinn, 2005

Does not meet selection criteria (does not answer research question, compares center protocols, not surgical techniques).

Flores, 2008

Does not meet selection criteria (none of the relevant outcome measures reported)

Gilleard, 2014

Included RCT of this review is already described in previous version of this guideline

Grobbelaar, 1994

Does not meet selection criteria (does not answer research question).

Grobbelaar, 1995

Excluded on study design (observational study)

Halli, 2012

Excluded on study design (observational study)

Hardwicke, 2014

Descriptive review included studies before 2014

Hassan, 2005

Does not meet selection criteria (follow-up too short)

Hassan, 2007

Excluded on study design (observational study)

Holtmann, 1984

Does not meet selection criteria (follow-up too short)

Kappen, 2018

Review of cohort and cross-sectional studies

Karling, 1998

Does not meet selection criteria (does not answer research question).

Kitagawa, 2003

Does not meet selection criteria (palatum durum)

Latham, 2007

Does not meet selection criteria (more of a timing than technique issue)

Lee, 2013

Does not meet selection criteria (palatum durum)

Leenstra, 1996

Does not meet selection criteria (follow-up too short)

Maggiuli, 2014

Does not meet selection criteria (outcome: maxillary growth, follow-up: 6 months postoperatively; too short to draw conclusions regarding maxillary growth)

McWilliams, 1995

Excluded on study design (observational study)

Meazzini, 2008

Does not meet selection criteria (does not answer research question, compares center protocols, not surgical techniques).

Miachon, 2014

Narrative review

Minatel, 2019

Review included only 1 RCT (Ganesh, 2015) which is described in the guideline

Mǿlsted, 1993

Does not meet selection criteria (palatum durum)

Nasser, 2008

Does not meet selection criteria (palatum durum)

Nollet, 2007

Does not meet selection criteria (does not answer research question, compares center protocols, not surgical techniques).

Reddy, 2010

Excluded on study design (observational study)

Rose, 2001

Does not meet selection criteria (none of the relevant outcome measures reported)

Rossell-Perry, 2014

Does not meet selection criteria (describes uvular repair, not palate repair)

Shaw, 1992

Does not meet selection criteria (not an original article)

Sommerlad, 2001

Does not meet selection criteria (does not answer research question, does not compare different surgical techniques).

Stein, 2019

Included RCTs in this review are already described in previous version of this guideline

Syafrudin Hak, 2011

Does not meet selection criteria (compares orthopaedic treatment not surgical techniques)

Tahir, 2017

RCT does not meet selection criteria (follow-up too short)

Tanino, 1997

Number of participants too low (<10 per group)

Timbang, 2014

Included RCT of this review is already described in previous version of this guideline

Trotman, 1996

Does not meet selection criteria (palatum durum)

Wada, 1990

Does not meet selection criteria (palatum durum)

Wermker, 2013

Does not meet selection criteria (study includes adult patients)

Witt, 1999

Excluded on study design (observational study)

Autorisatiedatum en geldigheid

Laatst beoordeeld  : 26-11-2021

Laatst geautoriseerd  : 26-11-2021

The Board of the Dutch Society for Plastic and Reconstructive Surgery (NVPC) will assess whether this guideline module is still up-to-date in 2026 at the latest. If necessary, a new working groupwill be appointed to revise the guideline module. The validity of the guideline or modules of the guideline may lapse earlier when new developments arise. As the holder of this guideline, the NVPC is chiefly responsible for keeping the guideline up to date.

 

Module1

Responsible party2

Year of autorisation

Next assessment of actuality guideline3

Frequency of assessement of actuality4

Supervisor of actuality5

Relevant factors for changes in recommendations6

Technique repairing cleft lip, alveolus and/or palate

NVPC

2021

2026

every 5 years

NVPC

None

[1] Name of module

2 Responsible party for the module

3 maximum of 5 years

4 half a year, every (other,..) year

5 supervising party or parties

6 Current reseach, changes in organizations/restitions, new available rescourses

 

Other scientific organizations participating in the guideline or users of the guideline share the responsibility to inform the chiefly responsible party (NVPC) about relevant developments within their fields.

Initiatief en autorisatie

Initiatief:
  • Nederlandse Vereniging voor Plastische Chirurgie
Geautoriseerd door:
  • Nederlandse Vereniging voor Keel-Neus-Oorheelkunde en Heelkunde van het Hoofd-Halsgebied
  • Nederlandse Vereniging voor Kindergeneeskunde
  • Nederlandse Vereniging voor Obstetrie en Gynaecologie
  • Nederlandse Vereniging voor Plastische Chirurgie
  • Vereniging Klinische Genetica Nederland
  • Nederlands Instituut van Psychologen
  • Nederlandse Vereniging voor Mond- Kaak- en Aangezichtschirurgie
  • Nederlandse Verenging voor Schisis en Cranio Faciale Afwijkingen
  • Nederlandse Vereniging van Orthodontisten

Algemene gegevens

The revision of this guideline module was supported by Knowledge Institute Federation of Medical Specialists (https://www.demedischspecialist.nl/kennisinstituut) and was financed by the Quality Foundation of the Dutch Medical Specialists (SKMS). The funding organization did not have any influence on the content of the guideline in any way.

Doel en doelgroep

Objectives of the guideline

The aim of this guideline is to improve the care of children/patients with CLA/Ps in The Netherlands ranging from prenatal detection to young adulthood, substantiated by scientific knowledge from research where possible. ‘Improving’ also means providing insight in the differences in practices between cleft teams and discriminating between wanted and unwanted (i.e. scientifically based or non-scientifically based) practice variation. This resulted in recommentations for a more uniform treatment. However, the lack of high-quality studies and evidence remains a serious limiting factor and forced the working group to define some conclusions in a more generalized way than was wished for at the start.

 

Specific attention will be given to the following aspects:

  1. reducing undesirable/unfounded practice variation in the working method and treatment protocols of the Dutch cleft teams, without hampering custom work, innovation or research;
  2. making objective / evidence-based information about the treatment of CLA/Ps available and accessible to healthcare providers, patients, parents and other parties;
  3. determine to what extent the existing organisation of care needs to be changed in order to meet the requirements regarding “state of the art” treatment of a child or adult with CLA/Psand the follow-up to this treatment.

 

In this manner, the guideline offers a tool to create more uniform care in the field of the prenatal and postnatal treatment of a child with CLA/Ps and the implementation of this care in the Netherlands.

 

Intended users of the guideline

The guideline is primarily intended for all healthcare professionals who are involved in caring for a child with CLA/Ps: general practitioners, midwives, gynaecologists, paediatricians, ENT physicians, plastic surgeons, maxillofacial surgeons, orthodontists, clinical geneticists, specialised nurses, speech therapists, (paediatric) dentists, medical psychologists, remedial educationalists and social workers. The secondary target group involves patients, parents and their surroundings.

Samenstelling werkgroep

A multidisciplinary working group was appointed by the Dutch Society for Plastic and Reconstructive Surgery in October 2019 to update the existing guidelines for clefts of the lip and palate. The original guidelines were initiated by the Dutch Society for Plastic and Reconstructive Surgery and this Society remains responsible for the revisions. The working group subsequently updated both the guideline for prenatal counsellling for clefts of the lip, alveolus, and/or palate (Counseling na prenataal vastgestelde schisis, 2011) and postnatal treatment (Behandeling van patiënten met een schisis, 2018). The working group consisted of representatives from all relevant specialties involved in the care for patients with cleft lip, alveolus and/or palate. Members were mandated by their professional organizations. The working group consisted of a mix of new members and members, who worked on previous editions as well. The group worked on the update of the guideline for two years. The working group is responsible for the full text of this guideline.

 

Working group

  • Dr. A.B. Mink van der Molen, MD, plastic surgeon, Universitair Medisch Centrum Utrecht, (chairman), NVPC
  • Dr. M.F. van Dooren, clinical geneticist, Erasmus MC Rotterdam, VKGN
  • Dr. M.J.H. van den Boogaard, clinical geneticist, Universitair Medisch Centrum Utrecht, VKGN
  • Dr. L.N.A. van Adrichem, MD, plastic surgeon, Universitair Medisch Centrum Utrecht, NVPC
  • Dr. H.F.N. Swanenburg de Veye, psychologist, Universitair Medisch Centrum Utrecht/Wilhelmina Kinderziekenhuis, Utrecht, NIP
  • Dr. C.J. Bax, MD, gynaecologist, Amsterdam UMC, NVOG
  • Prof. dr. C.C. Breugem, MD, plastic surgeon, Amsterdam Medical Center and Meander Medical Center, NVSCA
  • Drs. F. Bierenbroodspot, MD, Oral and maxillofacial surgeon, Isala, Zwolle, NVMKA
  • Drs. M. Haasnoot, MD, paediatrician, Wilhelmina Kinderziekenhuis, Utrecht, NVK
  • Drs. H.H.W. de Gier, MD, otolaryngologist, Erasmus MC Rotterdam, NVKNO
  • Dr. M.A.R Kuijpers, orthodontist, Radboud University Medical Center, NVvO
  • Dr. M.E.L. Nienhuijs, MD, Oral and maxillofacial surgeon, Radboud University Medical Center, NVSCA
  • Dr. D. de Haan, patient representative, Schisis Nederland

 

Advisory board

  • Drs. B. Spaan, dentist, CBT Vogellanden Zwolle, NVvK
  • I. Noureldin - Hop, orthopedagogue, NSDSK, NVO
  • M. J. Coerts, speech therapist, Amsterdam UMC, NVLF

 

With methodological support of

  • Drs. A.A. Lamberts, senior advisor, Knowledge Institute Federation of Medical Specialists
  • Dr. M. den Ouden - Vierwind, advisor, Knowledge Institute Federation of Medical Specialists

Belangenverklaringen

According to the KNMG-code, all members of the working group have declared in writing if, in the last five years, they have held a financially supported position with commercial businesses, organisations or institutions that may have a connection with the subject of the guidelines. Enquiries have also been made into personal financial interests, interests pertaining to personal relationships, interests pertaining to reputation management, interests pertaining to externally financed research, and interests pertaining to valorisation of knowledge. These Declarations of Interest can be requested from the secretariat of the Knowledge Institute of Medical Specialists. See below for an overview.

 

Member

Profession

Side jobs

Declared conflicting interests

Actions

Dr. A.B. Mink van der Molen

plastic surgeon,

None

None

No actions

Dr. M.F. van Dooren

clinical geneticist

Co-chair VKGN

None

No actions

Dr. M.J.H. van den Boogaard

clinical geneticist

None

None

No actions

Dr. L.N.A. van Adrichem

plastic surgeon

DGA van Adrichem Medical B.V.

Chairman Concilium plastico chirurgicum

Member Raad Opleiding

Member BBC NVPC

Advisor Hoofdmaatje

Chairman Medical Council Equipe Zorgbedrijven

Member Medicatie Commissie Equipe Zorgbedrijven

Member stuurgroep STW project TU-Twente

None

No actions

Dr. H.F.N. Swanenburg de Veye

psychologist

None

None

No actions

Dr. C.J. Bax

gynaecologist

Volunteer hospice

Member NIPT consortium

Member committee quality documents NVOG

Secretary committee Otterlo NVOG

Treasurer working group infectious diseases NVOG

None

No actions

Dr. C.C. Breugem

plastic surgeon

None

None

No actions

Drs. F. Bierenbroodspot

Oral and maxillofacial surgeon

Working Group Esthetische Aangezichtschirurgie

None

No actions

Drs. M. Haasnoot

paediatrician

None

None

No actions

Dr. J. de Gier

otolaryngologist

Board member NVSCA

None

No actions

Dr. M. Kuijpers

orthodontist

Guideline committee Mondzorg voor jeugdigen preventie diagnostiek behandeling

None

No actions

Dr. M. Nienhuijs

Oral and maxillofacial surgeon

Boardmember NVSCA

None

No actions

Dr. D. de Haan

patient representative,

Teacher/ education advisor HU-PABO, Hogeschool Utrecht

None

No actions

Inbreng patiëntenperspectief

Patients were represented by Schisis Nederland. Schisis Nederland is an independent organization representing patients with CLA/Psand their parents in the Netherlands. Representatives from Schisis Nederland participated in the working group. The concept guideline module was presented to Schisis Nederland for their comments.

 

Qualitative estimation of possible financial impact under the Wkkgz / Kwalitatieve raming van mogelijke financiële gevolgen in het kader van de Wkkgz

In accordance with the Wet kwaliteit, klachten en geschillen zorg (Wkkgz), a qualitative estimation has been made whether the recommendations in the guideline may lead to substantial financial consequences. In carrying out this assessment, guideline modules were tested in various domains (see the flow chart).

The qualitative estimate shows that there is probably no substantial financial impact, see table below.

 

Module

Results qualitative estimation

Elucidation

Module Techniek lip- en palatumsluiting bij patiënten met een schisis

Likely no substantial financial impact

The recommendation(s) are not widely applicable (<5,000 patients) and are therefore expected to have no substantial financial consequences for public expenditure.

Methode ontwikkeling

Evidence based

Implementatie

Guideline implementation and practical applicability of the recommendations was taken into consideration during various stages of guideline development. Factors that may promote or hinder implementation of the guideline in daily practice were given specific attention. The guideline is distributed digitally among all relevant professional groups. The guideline can also be downloaded from the Dutch Society for Plastic and Reconstructive Surgery website: www.nvpc.nl, and the guideline website: www.richtlijnendatabase.nl. The implementation table can be found in the related products.

Werkwijze

AGREE

The guideline has been drafted in accordance with the requirements outlined in the ‘Guidelines 2.0’ report of the Guideline Advisory Committee of the Council on Science, Education and Quality (WOK). This report is based on the AGREE II instrument (Appraisal of Guidelines for Research & Evaluation II) (Brouwers, 2010), an instrument designed to assess the quality of guidelines with broad international support (Brouwers, 2010). The development of a evidence-based guideline module is described step-by-step in “Ontwikkeling van Medisch Specialistische Richtlijnen” of Knowledge Institute for Medical Specialists.

 

Inventory of the problem areas

During the preparation phase the working group used an inventory to find the problem areas. A report of this inventory can be found in the related products.

 

Primary questions and outcome measures

Based on the outcomes of the bottleneck analysis, the president and advisor formulated draft primary questions. These were discussed and defined together with the working group. Subsequently, the working groupdetermined which outcome measures were relevant for the patient for each primary question, examining both desired and undesirable effects. The working groupvaluated these outcomes based on their relative importance as crucial, important and unimportant.

 

Literature search and selection strategy

Specific search terms were used to identify published scientific studies related to each individual primary question in electronic databases like Medline, Cochrane, and Embase. Additionally, the references of the selected articles were screened for additional relevant studies. Studies offering the highest level of evidence were sought out first. Members of the working group selected articles identified by the search based on predetermined criteria. Theselected articles were used to answer the primary question. The searched databases, the search string or terms used during the search and selection criteria applied are listed in the module for each individual primary question.

 

Quality assessment of individual studies

Individual studies were assessed systematically based on predefined methodological quality criteria in order to assess the risk of biased study results. These assessments may be found in the column ‘Study quality assessment’ in an evidence table.

  • AMSTAR - for systematic reviews.
  • Cochrane - for randomized controlled trials.
  • ACROBAT-NRS - for observational studies.
  • QUADAS II - for diagnostic studies.

 

Summary of the literature

The relevant study results from all selected articles were presented clearly in evidence tables. The key findings from the literature are described in the literature summary. If studies were sufficiently similar in design, data were also summarized quantitatively (meta-analysis) using Review Manager 5.

 

Assessment of the level of scientific evidence

With regard to intervention questions, the level of scientific evidence was determined using the GRADE method. GRADE is short for ‘Grading Recommendations Assessment, Development and Evaluation’ (see http://www.gradeworkinggroup.org/)

 

GRADE distinguishes four grades of quality of evidence, i.e. high, moderate, low and very low. These grades indicate the degree of confidence in the conclusions in the literature ((Schünemann, 2013; Hultcrantz, 2017).

 

GRADE

Definition

High

  • There is a high degree of confidence that the true effect of treatment is very close to the estimated effect of treatment as reported in the conclusion in the literature.
  • It is very unlikely that the conclusion drawn in the literature will change if further research is done.

Moderate

  • There is a moderate degree of confidence that the true effect of treatment is very close to the estimated effect of treatment as reported in the conclusion in the literature.
  • It is possible that the conclusion drawn in the literature will change if further research is done.

Low

  • There is a limited degree of confidence that the true effect of treatment is very close to the estimated effect of treatment as reported in the conclusion in the literature.
  • It is probable that the conclusion drawn in the literature will change if further research is done.

Very low

  • There is little confidence that the true effect of treatment is very close to the estimated effect of treatment as reported in the conclusion in the literature.
  • The conclusion is very uncertain

 

According to the GRADE methodology the clinical decision threshold should play an important role in assessing the level of evidence (grading) in guidelines (Hultcrantz, 2017). To set the threshold all critical outcomes, and the considerations should be determined. The clinical decision threshold is not exactly the same as the Minimal Clinically Important Difference (MCID). In situations in which an intervention has no important disadvantages and low costs, the clinical decision threshold with regard to the efficiency of an intervention can be lower (closer to zero/ no effect) than MCID (Hultcrantz, 2017).

 

Formulation of conclusions

For interventions, the conclusion does not refer to one or more articles, but is drawn based on the body of evidence. The working group looked at the net benefits of each intervention. This was done by determining the balance between favourable and unfavourable effects for the patient.

 

With regard to questions about the value of diagnostic tests, harm or adverse effects, aetiology and prognosis, the scientific evidence is summarized in one or more conclusions, listing the level of evidence for the most relevant data.

 

Considerations

When making recommendations, scientific evidence was considered together with other key aspects, such as expertise of the group members, patient preferences, costs, availability of facilities and/or organizational aspects. Insofar as they are not part of the systematic literature review, these aspects are listed under ‘Considerations’. The considerations are written using a structured format based on the evidence-to-decision framework of the international GRADE Working Group, and part of the GRADE methodology (Alonso-Coello, 2016a; Alonso-Coello 2016b).

 

Formulation of recommendations

Recommendations provide an answer to the primary question and are based on the best scientific evidence available and the most important considerations. The level of scientific evidence and the importance given to considerations by the working group jointly determine the strength of the recommendation. In accordance with the GRADE method, a low level of evidence for conclusions in the systematic literature review does not rule out a strong recommendation, while a high level of evidence may be accompanied by weak recommendations (Agoritsas, 2017; Neumann, 2016). The strength of the recommendation is always determined by weighing all relevant arguments.

 

Preconditions (Organisation of care)

In the analysis of problem areas, the organisation of care (all those aspects that are preconditions for the provision of care) were explicitly taken into account. These aspects include coordination, communication, materials, financial means, work force and infrastructure. Preconditions that are relevant to the answering of a specific clinical question are part of the considerations related to that specific question.

 

Knowledge gaps

During the development of this guideline, systematic searches were conducted for research contributing to answering the primary questions. For each primary question, the working group determined whether (additional) scientific research is desirable.

 

Commentary and authorization phase

The draft guideline was submitted to the (scientific) organizations involved for comment. The guideline was also submitted to the following organizations for comment: Dutch College of General Practitioners (NHG), Healthcare Insurers Netherlands (ZN), The Dutch Healthcare Authority (NZA), the National Health Care Institute (ZINL), the Health Care Inspectorate (IGJ), Dutch Organisation of Hospitals (NVZ), Dutch Federation of Academic Hospitals (NFU), Dutch Organisation of Independent Clinics (ZKN), the Netherlands Patients Federation, Dutch Organisation of nurses and caregivers (V&VN), Dutch Association of Physician Assistants, and Collaborating Top Clinical Training Hospitals (STZ). Comments were collected and discussed with the working group. The draft guideline was updated and finalized by the working group based on the comments. The final guideline was submitted for authorization to the (scientific) organizations involved and authorized or approved by them.

 

Literature

Agoritsas T, Merglen A, Heen AF, Kristiansen A, Neumann I, Brito JP, Brignardello-Petersen R, Alexander PE, Rind DM, Vandvik PO, Guyatt GH. UpToDate adherence to GRADE criteria for strong recommendations: an analytical survey. BMJ Open. 2017 Nov 16;7(11):e018593. doi: 10.1136/bmjopen-2017-018593. PubMed PMID: 29150475; PubMed Central PMCID: PMC5701989.

Alonso-Coello P, Schünemann HJ, Moberg J, Brignardello-Petersen R, Akl EA, Davoli M, Treweek S, Mustafa RA, Rada G, Rosenbaum S, Morelli A, Guyatt GH, Oxman AD; GRADE Working Group. GRADE Evidence to Decision (EtD) frameworks: a systematic and transparent approach to making well informed healthcare choices. 1: Introduction. BMJ. 2016 Jun 28;353:i2016. doi: 10.1136/bmj.i2016. PubMed PMID: 27353417.

Alonso-Coello P, Oxman AD, Moberg J, Brignardello-Petersen R, Akl EA, Davoli M, Treweek S, Mustafa RA, Vandvik PO, Meerpohl J, Guyatt GH, Schünemann HJ; GRADE Working Group. GRADE Evidence to Decision (EtD) frameworks: a systematic and transparent approach to making well informed healthcare choices. 2: Clinical practice guidelines. BMJ. 2016 Jun 30;353:i2089. doi: 10.1136/bmj.i2089. PubMed PMID: 27365494.

Brouwers MC, Kho ME, Browman GP, et al. AGREE Next Steps Consortium. AGREE II: advancing guideline development, reporting and evaluation in health care. CMAJ. 2010;182(18):E839-42. doi: 10.1503/cmaj.090449. Epub 2010 Jul 5. Review. PubMed PMID: 20603348.

Hultcrantz M, Rind D, Akl EA, et al. The GRADE Working Group clarifies the construct of certainty of evidence. J Clin Epidemiol. 2017 Jul;87:4-13. doi: 10.1016/j.jclinepi.2017.05.006. Epub 2017 May 18. PubMed PMID: 28529184.

Medisch Specialistische Richtlijnen 2.0 (2012). Adviescommissie Richtlijnen van de Raad Kwalitieit. http://richtlijnendatabase.nl/over_deze_site/over_richtlijnontwikkeling.html.

Schünemann H, Brożek J, Guyatt G, et al. GRADE handbook for grading quality of evidence and strength of recommendations. Updated October 2013. The GRADE Working Group, 2013. Available from http://gdt.guidelinedevelopment.org/central_prod/_design/client/handbook/handbook.html.

Schünemann HJ, Oxman AD, Brozek J, et al. Grading quality of evidence and strength of recommendations for diagnostic tests and strategies. BMJ. 2008;336(7653):1106-10. doi: 10.1136/bmj.39500.677199.AE. Erratum in: BMJ. 2008;336(7654). doi: 10.1136/bmj.a139. PubMed PMID: 18483053.

Wessels M, Hielkema L, van der Weijden T. How to identify existing literature on patients' knowledge, views, and values: the development of a validated search filter. J Med Libr Assoc. 2016 Oct;104(4):320-324. PubMed PMID: 27822157; PubMed Central PMCID: PMC5079497.

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