or cleft palate: Middle ear findings and hearing during childhood

International Journal of Pediatric Otorhinolaryngology 111 (2018) 47–53

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Internationally adopted children with cleft lip and/or cleft palate: Middle ear findings and hearing during childhood☆

T

C.L. Werker∗, M.T.A. van den Aardweg, S. Coenraad, A.B. Mink van der Molen, C.C. Breugem Department of Plastic Surgery and Department of Otolaryngology, Wilhelmina Children's Hospital, University Medical Centre Utrecht, Lundlaan 6, 3584 EA, Utrecht, The Netherlands

A R T I C LE I N FO

A B S T R A C T

Keywords: Cleft lip and/or cleft palate Otitis media with effusion Ventilation tubes Hearing

Objective: Adopted children with cleft lip and/or cleft palate form a diverse group of patients. Due to increased age at palatal repair, adopted children have a higher risk of velopharyngeal insuffiency and poor speech outcome. Delayed palate repair may also lead to longer lasting Eustachian tube dysfunction. Decreased function of the Eustachian tube causes otitis media with effusion and recurrent acute otitis media, which can lead to other middle ear problems and hearing loss. Methods: One-hundred-and-thirty-two adopted children treated by the Cleft palate team in Wilhelmina Children's Hospital during January 1994 and December 2014 were included. Retrospectively, middle ear findings, the need for ventilation tube insertion and hearing during childhood were assessed. Findings were compared with 132 locally born children with cleft lip and/or cleft palate. Results: Adopted children had a mean age of 26.5 months old when they arrived in our country. After the age of two the total number of otitis media with effusion episodes and the need for ventilation tube placement did not significantly differ among adopted and non-adopted children. Adopted children had significantly more tympanic membrane perforations. Hearing threshold levels normalized with increasing age. Although within normal range, adopted children showed significantly higher pure tone averages than locally born children when they were eight to ten years old. Conclusion: In general, adopted patients with cleft lip and/or cleft palate did not have more middle ear problems or ventilation tubes during childhood. However, theyhave more tympanic membrane perforations.

1. Introduction Overall adoption numbers in the Netherlands have declined over the past years, but the number of adopted children with ‘special needs’ has increased [1]. ‘Special needs’ children are children with congenital malformations, such as cleft lip with or without cleft palate and congenital hand and feet defects [1,2]. Of all 214 internationally adopted children in the Netherlands in 2016 approximately 80% had a medical condition or birth defect. Cleft lip and/or cleft palate (CLP) is common [1]. Consequently our cleft palate team is confronted frequently with the care for these children. Cleft care for internationally adopted children with CLP varies between institutions in the Netherlands due to insufficient evidence to guide treatment. Institutions that described their patient characteristics and initial care and treatment demonstrate a very diverse group of patients who often have velopharyngeal insuffiency and poor speech outcome [2–7]. Different factors are thought to contribute such as a) the new child-family relationship, b) palate and

☆ ∗

Work presented at: Dutch Cleft Palate Association meeting November 18 2017. Corresponding author. E-mail address: [email protected] (C.L. Werker).

https://doi.org/10.1016/j.ijporl.2018.05.019 Received 9 March 2018; Received in revised form 15 May 2018; Accepted 17 May 2018 Available online 19 May 2018 0165-5876/ © 2018 Published by Elsevier B.V.

lip repair at an older age with more risks of complications [8,9], c) the variety and quality of former treatments [2–6] with possibly re-do surgery and d) having to learn a new language. Furthermore, children with CLP often suffer from conductive hearing loss due to otitis media with effusion (OME) and recurrent acute otitis media (AOM) [10–13]. Repair of the cleft palate should lead to a more normal Eustachian tube function and thus to a decrease of OME [14–17]. Since palatoplasty in adopted children with CLP commonly takes place at an older age, they have longer exposure to OME. Current Dutch guidelines recommend insertion of ventilation tubes in children with recurrent acute otitis media, defined as three episodes of AOM per six months or four episodes per year, and in children with persistent otitis media with effusion defined as, OME persisting for more than 3 months with a conductive hearing loss of 25 dB or more [20–25]. Nevertheless, the benefits of ventilation tubes are still subject of debate as in some cases the placement of ventilation tubes itself leads to complications, such as tympanic membrane perforations and

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2.2. Palatoplasty

otorrhea [18,26]. Otorrhea particularly becomes a problem when it is caused by resistant organisms, such as Methicillin-resistant Staphylococcus aureus (MRSA). [27] Internationally adopted children are often adopted from countries with a high MRSA carrier rate, they have an increased risk of MRSA-colonization and therefore an increased risk of MRSA otorrhea [2,28–30]. The precise effect of MRSA on the rate of postoperative infections and complications after palate repair and ventilation tube insertion is unknown [2]. However, in our institution otolaryngologists are reluctant to place ventilation tubes in children carrying MRSA because of the fear for chronic otorrhea and the subsequent inability to eradicate MRSA. Although some studies have been published about speech assessment [2–7] in the adopted cleft palate patients, none have investigated middle ear and hearing problems in this group. Therefore, the aim of this study was to describe characteristics of adopted children with CLP and compare middle ear findings and hearing in adopted children with CLP with locally born children with CLP during childhood in the Netherlands.

During study period different plastic surgeons performed palatoplasty. Surgical techniques used in adopted children who had palatoplasty in their native country were unknown. Adopted and non-adopted children that underwent palatoplasty in our hospital before the year of 2007 (30%), were likely to have a palatoplasty according to Furlow's technique with double opposing Z-plasty [62]. Children that underwent surgery after the year of 2007 (70%) underwent an intravelar veloplasty under a microscope following Sommerlad's principles [63]. 2.3. Otolaryngology Every consultation to the otolaryngologist ideally consisted of otoscopic examination, tympanometry and audiological examination. However, due to MRSA in the adopted group and uncooperativeness of patients not all children underwent all these examinations and tests during every visit.

2. Patients and methods

2.3.1. Tympanometry Tympanometry was performed by an audiologist using an Interacoutics® impedance audiometer AT235h with a 226-Hz probe. Each tympanogram was scored according to the Jerger classification into either subtype A, B, C or E and judged by the otolaryngologist as either normal or abnormal. Subtype A was considered to be normal, whereas subtypes B, C and E were considered abnormal [61].

2.1. Study population A retrospective study including all internationally adopted children with CLP presenting to the cleft palate team outpatient clinic in the Wilhelmina Children's Hospital Utrecht between January 1994 and December 2014 was performed. One-hundred-and-two patients were identified. To make comparison possible, we included a control group consisting of non-adopted CLP children also treated by the cleft palate team in Wilhelmina Children's hospital. To improve the efficiency of our analysis we applied frequency matching to select our controls. Locally born controls were matched for gender and cleft type with the adopted group. These two characteristics were chosen for matching because the distribution of both gender and cleft type in our adopted group differs from what is to be expected in the general population. In the general cleft population 50% of the population suffers from cleft lip and palate, 30% has a cleft palate and 20% has cleft lip [31]. As a result of frequency matching 132 non-adopted children born between January 1997 and December 2011 were randomly selected. Medical records of all included patients were reviewed, information concerning surgical and medical history were obtained. Study data were collected and managed in a database. The cleft palate team encompasses various specialties including pediatrics, plastic surgery, otolaryngology, audiology, speech and language therapy, dentistry and maxillo-facial surgery, leading to multidisciplinary treatment. Although internationally there are still many unresolved questions about timing in cleft palate closure, our cleft palate team did use a standard schedule with minimal consultations for locally born children (see Table A in the appendix). However, many patients consulted specific specialists more frequently. For adopted children this schedule was often not applicable, since they arrived in the Netherlands at an older age. For these children treatment often started with a consultation by a general pediatrician for a thorough medical history and physical examination. If the MRSA status was unknown, tests were done. When a patient tested positive, attempts to eradicate MRSA were often postponed until after the palatal closure was performed, as is advised in our hospital protocol [32]. In our adoption group 36 children (27%) tested MRSA positive when they first arrived. After examination by the general pediatrician, a referral to the cleft team followed. Depending on the treatment the adopted children underwent in their country of origin and the MRSA status the adopted children saw various specialists at their initial presentation to the team. The plastic surgeon was consulted to see whether (re-do) surgery was necessary. An otolaryngologist examined the children to determine if myringotomy or ventilation tube insertion was required.

2.3.2. Audiometry Pure tone audiometry was performed with Decos audiometer in a soundproof room. Ear specific measurements were obtained at 250 Hz, 500 Hz, 1000 Hz, 2000 Hz and 4000 Hz and registered in dB hearing level by trained audiologists using headphones. In younger children, aged six months to thirty months, behavioral observational audiometry was used to assess hearing. To compare different audiometry results, pure tone averages were calculated. Hearing was considered normal with mean threshold levels of 0–20 dB. Mild hearing loss was defined as mean hearing threshold levels between 21 and 40dB. If thresholds were higher than 40 dB it was considered as severe hearing loss. 2.4. Statistical analyses For statistical analysis SPSS version 22.0 was used. Demographic and surgical data were analyzed with descriptive statistical methods. Normally distributed data outcomes were compared using chi-square test for categorical and dichotomous data and t-test for continuous data. Results were presented as percentages in categorical and dichotomous data and as means with standard deviation in continuous data. To compare outcomes in not normally distributed data a Mann-Whitney test was used. Results were presented as median with a range. Correlations between different variables were calculated using Pearson's correlation test for normally distributed and continuous data and Spearman's correlation test for not normally distributed data and for categorical and dichotomous data. Data concerning OME episodes, ventilation tube insertion and hearing loss were analyzed in total and for different age groups; zero to two years old, two to four years old, four to six years old, six to eight years old, eight to ten years old and older than ten years old. Negative binomial regression analysis was used to find predictors in count data. Linear regression analysis was used to find predictors in continuous data. A p-value of less than 0.05 was considered statistically significant. 2.5. Ethics The medical ethical review board of our institution approved this study (16–656). The requirement for informed consent was waived. 48

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Table 1 Adoptees compared to non-adoptees concerning demographic and surgical characteristics. Adoption (N = 132)

Gender, n (%) Boys Girls Age at time of the study: Median (range); months Cleft type, n (%) Cleft lip/cleft alveolus Unilateral cleft lip and palate Bilateral cleft lip and palate Cleft palate Age cleft lip repair: Median (range); months One-stage vs two stage repair, n (%) One stage Two stage Mean age soft palate repair: Median (range); months Mean age hard palate repair: Mean (SD); months a b c

Non-adoption (N = 132)

p

Mean difference

95% confidence interval Lower

Upper

90 (68.2) 42 (31.8) 122 (39–281)

90 (68.2) 42 (31.8) 116 (42–233)

–

–

–

–

61b

–

–

–

20 (15.2) 66 (50.0) 37 (28.0) 9 (6.8) 10.0 (1–67)

20 (15.2) 66 (50.0) 37 (28.0) 9 (6.8) 4.0 (1–12)

–

–

–

–

00b

–

–

–

78 (69.6) 34 (30.4) 25.0 (5–54) 61.1 (17.7)

69 (61.6) 43 (38.4) 10.0 (3–34) 58.9 (22.5)

21c

–

–

–

– 2.16

– −7.09

– 11.40

b

00 64a

Independent samples t-test. Mann-Whitney test. Chi-square test.

3. Results

Table 2 Otological findings during study period in total study population, adoptees compared to non-adoptees.

3.1. Demographics The 132 adopted patients with CLP consisted of 90 boys (68%) and 42 girls (32%). The mean age at adoption was 26.5 months old with a standard deviation of 14.3. Adoptees originated from Asia (93%), America (4%), Europe (2%) and Africa (1%). In most cases China was the country of origin, 118 patients (89%) were adopted from this country. One-hundred-and-four children (79%) underwent surgery in their country of origin. Seventy-four children (56%) had lip repair, 3 children (2%) had only palatal repair and 27 children (21%) had lip and palatal repair in their country of origin. The control group of non-adopted children also consisted of 132 children, the distribution of cleft type and gender was by definition similar to that in the adoption group. The distribution is shown in Table 1. No significant differences were found for age at the time of the study, age at hard palate closure and the method used to repair the palate (one-versus two-stage closure). However, compared to locally born children, adopted children were significantly older when they underwent lip repair and soft palate repair, see Table 1.

Otitis media with effusion n (%) Ventilation tubes n (%) Otorrhea n (%) Atelectasis or retraction of the tympanic membrane n (%) Perforation of the tympanic membrane n (%) Cholesteatoma n (%) a

Total (N = 247)

Adoption (N = 120)

Non-adoption (N = 127)

p

202 (81.8) 164 (66.4) 87 (36.3) 37 (15.0)

92 75 37 16

110 (86.6) 89 (70.1) 50 (40.0) 21 (16.5)

.04a .21a .21a .48a

47 (19.0)

29 (24.2)

18 (14.2)

.04a

2 (0.8)

2 (1.7)

0 (0.0)

.14a

(76.7) (62.5) (32.2) (13.3)

Chi-square test.

membrane perforations than non-adopted children, χ2 (1) = 4.29, p < 0.05. Otitis media with effusion was significantly more common in the non-adopted children, χ2 (1) = 4.10, p < 0.05. Chronic otorrhea occurred in 6.1% of the adopted children and 12.9% of the non-adopted children, without significant difference, χ2 (1) = 3.58, p = 0.06. Chronic otorrhea was defined as otorrhea lasting longer than 2 weeks. 3.4. Otitis media with effusion

3.2. Consultation Two-hundred-and-two children (82%) had at least one episode of otitis media with effusion. The number of observed episodes varied from one episode to twenty-two episodes per patient. OME was diagnosed 1263 times, 73% of these episodes were bilateral. A significantly lower number of OME episodes was found in adoptees (Median = 3.0) compared to non-adoptees, (Median = 5.0), U = 5346.5, z = −3.72, p < 0.01, r = −0.24. The mean or median number of OME episodes for different age categories is shown in Table 3.The only significant difference found is in the age group of 0–2 years old. The adopted group is very small compared to the non-adopted group, because adoption frequently took place after the age of two. If this age group is excluded from Mann-Whitney U testing, no significant difference in the total number of OME episodes during childhood is found between both groups, (Median = 3.0), U = 6.768.0, z = −1.11, p = 0.27, r = −0.07. Multivariable negative binomial regression analysis was performed to determine which available patient characteristics formed predictors for the total number of OME episodes after the age of two, see Table 4. The model was not significant (p = 0.44). None of the included

Of the 264 included children, 247 visited the otolaryngology department of our outpatient clinics at least once. The children that never visited our otolaryngologist were twelve children from the adopted group, ten children had cleft lip and/or alveolus and two had cleft lip and palate. The latter two children were both seen by the plastic surgeon of our cleft team once for second opinion. Because they did not receive any treatment in our hospital they were excluded from analysis. Of the locally born children six had never seen an otolaryngologist all with cleft lip and/or alveolus. The number of consultations ranged from 1 to 38 per person, with a median of eight visits. Non-adopted children (Median 9.0) had significantly more visits than adopted children (Median 7.0), U = 6224.0, z = −2.14, p = 0.03, r = −0.14. 3.3. Otological examination Table 2 shows the otological findings for both groups during the study period. Adopted children were more likely to develop tympanic 49

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Table 3 Mean or median episodes of otitis media with effusion for different age categories, adoptees compared to non-adoptees. Age, years

0–2 2–4 4–6 6–8 8–10 ≥10 Total a b

Adoption

Non-adoption

Mean difference

n

NV

Mean (SD)/Median (range)

n

NV

Mean (SD)/Median (range)

32 78 99 74 43 28 117

47 261 308 237 108 58 1014

1.50 (1.05) 2.09 (1.70) 1.55 (1.82) 0.0 (0–9) 0.0 (0–5) 0.0 (0–2) 3.0 (0–16)

88 97 99 81 45 24 126

288 345 313 215 108 70 1338

2.76 (1.61) 2.35 (2.23) 1.93 (1.82) 0.0 (0–7) 0.0 (0–6) 0.0 (0–3) 5.0 (0–22)

Lower

Upper

0.76 −0.33 -.0.14

1.76 0.85 0.91

p

.00a .38a .15a .95b .77b .35b .00b

Independent samples t-test. Mann-Whitney test. NV: Total number of visits during specific age period.

Table 4 Multivariable negative binomial regression analysis with patient characteristics as predictors of total number of OME episodes after the age of two years old (N = 204). Predictor

B

Adoption Gender Age at soft palate closure Cleft type UCLP BCLP

Incidence rate ratio

95% Wald Confidence interval Lower

Upper

Table 6 Multivariable negative binomial regression analysis with available patient characteristics as predictors of total number of ventilation tube insertions (N = 204).

p

−1.13 −0.43 0.02

0.33 0.65 1.02

0.05 0.15 0.93

2.17 2.78 1.11

.25 0.56 0.71

0.35 −0.66

0.52 1.42

0.14 0.05

14.35 5.49

0.77 0.58

Predictor

Adoption Gender Age at soft palate closure Cleft type UCLP BCLP Total OME episodes

*p < 0.05 is considered statistically significant, UCLP – unilateral cleft lip and palate, BCLP – bilateral cleft lip and palate.

A significant positive correlation was found between the total number of OME episodes and the total number of ventilation tubes placed, rs = 0.72, p < 0.001. A total number of 848 ventilation tubes were inserted. In the adoption group 75 children (63%) had one or more ventilation tube placements, whereas in locally born cleft patients 89 children (70%) needed at least one tube placement, the difference was not significant, χ2 (1) = 0.89, p = 0.35. The number of ventilation tube placements varied from 1 to 8 insertions per patient. The total number of ventilation tube insertions in adopted children with CLP (Median = 2.0) did not significantly differ from non-adopted children with CLP (Median = 4.0), U = 6488.5, z = −1.65, ns, r = −0.11. The median number of inserted ventilation tubes per age category is shown in Table 5. The only significant result is found in the age category zero to two years old, were adopted children (Median = 2.0) have

0–2 2–4 4–6 6–8 8–10 ≥10 Total

Non-Adoption (N = 127)

n

Median (range)

n

Median (range)

p

32 78 99 74 43 28 120

2.0 2.0 0.0 0.0 0.0 0.0 2.0

88 97 98 81 45 24 127

2.0 2.0 2.0 0.0 0.0 0.0 4.0

.04a .85a .30a .52a .78a .37a .10a

(0–4) (0–8) (0–6) (0–5) (0–3) (0–2) (0–16)

(0–6) (0–6) (0–6) (0–4) (0–4) (0–4) (0–18)

95% Wald Confidence interval Lower

Upper

p

0.08 0.11 0.01

1.09 1.11 1.01

0.66 0.75 0.99

1.80 1.64 1.03

0.75 0.60 0.49

−0.26 0.03 0.15

0.77 1.03 1.16

0.40 0.72 1.12

1.48 1.47 1.21

0.43 0.89 0.00*

3.6. Tympanometry Tympanometric results showed many missing values in both groups. Performed tympanogram mainly showed subtype B even after the age of ten. This is probably explained by the large number of OME episodes and ventilation tubes in children who underwent this examination.

Table 5 Median number of ventilation tubes inserted for different age categories adoptees compared to non-adoptees. Adoption (N = 120)

Incidence rate ratio

significantly less tubes inserted compared to locally born children (Median = 2.0), U = 1092.0, z = −2.11, p < 0.05, r = −0.19. Multivariable negative binomial regression analysis was performed to determine which available patient characteristics formed predictors for the total number ventilation tube insertions, see Table 6. The model was significant (p < 0.01). The total number of ventilation tube insertions was significantly associated with the total number of OME episodes, IRR 1.16, 95% CI 1.12–1.21, p < 0.01, a higher total number of OME episodes lead to an increase in the number of ventilation tubes inserted. No other significant predictors were found.

3.5. Ventilation tubes

Age, years

B

*p < 0.05 is considered statistically significant, UCLP – unilateral cleft lip and palate, BCLP – bilateral cleft lip and palate.

variables were predictors for the total number of OME episodes.

a

1.26 0.26 0.38

95% confidence interval

3.7. Hearing loss The number of audiometric examinations differed per patient, ranging from 1 to 25 examinations, with a median of 5 examinations per person. Pure tone averages for different age categories are shown in Table 7. Overall, hearing threshold levels normalize with increasing age. Although within normal range, adopted children had significantly higher pure tone averages than locally born children when they were eight to ten years old. No significant difference was found in other age categories, but at the age of eight to ten years old an increase in average hearing levels in seen in non-adopted children.

Mann-Whitney U test. 50

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recent study in the general population reported a percentage of 52% otorrhea, which is more similar to our findings [41]. Tympanic membrane perforations as complication after ventilation tube placement also occurred. Twenty-nine adoptees (25%) had at least one perforation, which was significantly more than the 18 children (14%) who suffered from this condition in the non-adopted group (p < 0.05). In patients with CLP tympanic membrane perforations occur in 1.3–10.6% of the ears treated with ventilation tubes [10,11,40,42–45]. The risk for perforation is associated with the type of ventilation tube inserted, the retention time and the number of tube insertions. In this study we did not find a higher number of tube insertions in the adoption, we were not able to specify the type of ventilation tubes inserted or the retention time. This may however have affected the tympanic membrane perforation rate in the adopted group. Further studies should be conducted. The most serious potential complication after ventilation tube insertion is the development of a cholesteatoma [18,46], but this risk is even higher in untreated ears with severe Eustachian tube dysfunction. Eustachian tube dysfunction with negative middle ear pressure may give rise to a retraction pocket followed by the development of a cholesteatoma [50]. Of all children with CLP 0.07–5.9% develops a cholesteatoma [47–51]. According to Djurhuus and colleagues (2015), patients with CLP and CP have a respectively 14-fold and 20-fold increased risk of surgically treated middle ear cholesteatoma at age 16 compared to random sample of the Danish population [47]. In our study population no cholesteatoma were found in the locally born patients, however two adoptees (1.7%) developed a cholesteatoma. Some cholesteatoma take a long time to develop [47], as a result there may be participants that will develop a cholesteatoma in the future. Conductive hearing loss during childhood caused by the mentioned middle ear problems, especially OME, in children with CLP is a common concept [12,20,52,53]. Hearing loss may be present within the first few months of life [53,54], whereas 18–20% of the children with isolated cleft lip and CLP fail their newborn hearing screening [55,56]. In our patient groups severe and mild hearing peaked in children aged zero to four years old, with increasing age the hearing threshold levels improved, but did not for all patients completely normalize as was described in other studies [37,57–59]. Decreased hearing levels at older age have been correlated with a greater number of ventilation tubes insertions [49]. In our study population, this correlation was not found. This may be explained by the fact that we only had a few patients that were older than ten years old at follow-up. Relevant literature on middle ear findings and hearing in cleft patients, as an outcome of cleft treatment, especially in adopted children, remains scarce. Future studies should further investigate the influence of late palate closure on middle ear status and hearing as the velopharyngeal muscles are also involved in Eustachian tube functioning [14,60]. Moreover, research should focus on the role of MRSA colonization in middle ear findings. Long-term and careful follow-up with special attention for this unique population is recommended.

Table 7 Pure Tone Average in different age categories adoptees compared to nonadoptees. Age, years

Adoption

Non-adoption

n

Mean (SD)

n

Mean (SD)

0–2

17

58

2–4

62

4–6

80

6–8

50

8–10

29

≥10

17

41.71 (9.06) 32.71 (12.13) 24.36 (9.87) 19.41 (8.37) 19.81 (8.52) 16.41 (10.22)

46.59 (13.01) 31.35 (12.06) 22.55 (9.86) 18.91 (8.92) 14.05 (7.19) 17.53 (7.57)

a

75 75 58 32 17

Mean difference

95% Confidence interval Lower Upper

p

4.89

−1.85

11.62

.15a

−1.36

−5.46

0.75

.52a

−1.82

−4.95

1.32

.25a

−0.51

−3.82

2.81

.76a

−5.76

−9.79

−1.74

.01a

1.12

−5.168

7.40

.72a

Independent samples t-test, SD – Standard deviation.

4. Discussion This study aimed to describe characteristics of adopted children with CLP and compare middle ear findings and hearing with locally born children with CLP during childhood. The adopted children mainly originated from China and arrived in the Netherlands around the age of two years old. Most children underwent lip repair in their country of origin, only a few underwent palatoplasty before adoption. Since in most children palatal closure was performed after adoption, it took place at a significantly older age compared to locally born children (p < 0.01). Due to increased age at time of primary palatal repair, a higher incidence of velopharyngeal insuffiency is found in adopted children [4,6]. Furthermore, late palatal closure may lead to more middle ear problems since it has been suggested that cleft palate leads to Eustachian tube dysfunction [15–17]. Based on the above, palatoplasty plays an important role in resolving OME and reducing the need for ventilation tube insertion. However, in a retrospective study by Nunn and colleagues (1995), very early cleft palate closure, within the first month of life, did not significantly reduce the need for ventilation tube placement in the first two-and-half years of life [33]. Moreover, our study showed a significant higher number of OME episodes in the non-adopted group compared to the adopted group, especially in the first two years of life (p < 0.01). Existing literature suggest that Chinese cleft patients have a lower rate of otological problems than Western population [34,35]. But since we only had information about the adopted children from the moment they arrived in the Netherlands, it seems likely that the number of OME episode in the adopted group during the first two years of life was underestimated. Especially since our results indicated that in other age categories the total number of OME episodes in both groups did not significantly differ. In locally born children OME was present within the first 6 months of life, it then peaked between the age of four and six years old, after which the number of episodes slowly decreased, but OME remained present even after the age of ten years old. These results were consistent with findings in previous studies [19,20]. In the non-cleft population the condition is most common below the age of two, but it has a bimodal distribution with a further peak at five years old [36]. OME is commonly treated with myringotomy and the insertion of a ventilation tube [39]. Nearly 70% of our study population had ventilation tubes inserted at least once, this is less than the 82.7% found in a recent Finnish study [38]. No significant difference was found between adopted and non-adopted children. A ventilation tube insertion can be associated with complications, such as otorrhea, tympanic membrane perforation and tympanosclerosis [37,39]. Otorrhea was found in 40% of the children in our study. Maheshwar and colleagues reported on persistent OME being present in 11.5% of their study population [40]. A

5. Limitations Some issues need further consideration. An important limitation was the retrospective nature of this study. Information in the medical records was often missing or incomplete. In addition, an underestimation of middle ear findings such as OME episodes and otorrhea episodes may have occurred, as patients may have gone to their general practitioner for treatment of their middle ear problems or to other regional hospitals to seek help. Furthermore, since no follow-up protocol is available, selection bias may influence the results, as patients in need of more intensive treatment were followed for a longer period. Moreover, loss to follow-up may have occurred as some children sought treatment in other hospitals due to the surgical waiting lists in our hospital. 51

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6. Conclusion

Funding

In general, adopted CLP patients did not have more middle ear problems or ventilation tubes during childhood. However, these children are known to experience velopharyngeal insuffiency and poor speech outcome and thus need extra attention. Further research is indicated.

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Conflicts of interest There are no conflicts of interest.

Appendix Table A . Protocol for outpatient clinic consultation and follow-up by cleft palate team in locally born children.

< 2 weeks after birth∗ 10 weeks∗ 4–6 months∗∗ 8–9 months∗∗ 2 years∗∗ 3 years 5 years 9 years∗∗∗ 15 years 18 years

Plastic surgeon

Orthodontist Oral Surgeon

Dentist Psychologist Clinical geneticist

x

x

x

x

x

x x

x

x

x x x x x

x x x x x

x x x

x x x x x

Speech pathologist

ENT specialist

Audiologist

x

x

x

x x x x x

x

∗

∗∗

x x x xˆ

xˆ

x

x

x

∗∗∗

x – Standard follow-up cleft lip and palate patients, ˆ - extra consultation, if necessary, CL, CLA, CLP, CP, CLP, CP, CLA, CLP. CL –Cleft lip, CLA – Cleft lip and alveolus, CP – Cleft palate, CLP – Cleft lip and palate. Surgery:Cheiloplasty – 3–4 months old, 6 weeks follow-upPalatoplasty – 6–12 months, 6 weeks follow-upAlveolar bone grafting – 9–11 years old, 6 weeks follow-up.

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