G Model
PEDOT-7326; No. of Pages 3 International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Difference in maxillary sinus volumes of patients with cleft lip and palate Gabriella Lopes de Rezende Barbosa a, Luiz Andre´ Pimenta b, Henrique Pretti c, Brent A. Golden d, Jason Roberts e, Amelia Fischer Drake e,* a
Departament of Oral Diagnosis, Division of Oral Radiology, Piracicaba Dental School, University of Campinas, Limeira Avenue, 901, Piracicaba, Sa˜o Paulo, Brazil Department of Dental Ecology, School of Dentistry, University of North Carolina at Chapel Hill, 001 Brauer Hall, Manning Dr & Columbia St. Chapel Hill, NC, USA c Department of Orthodontics, School of Dentistry, Federal University of Minas Gerais, Avenida Antoˆnio Carlos, 6627, Belo Horizonte, Minas Gerais, Brazil d Department of Oral & Maxillofacial Surgery, School of Dentistry, University of North Carolina at Chapel Hill, 149 Brauer Hall, Manning Dr & Columbia St. Chapel Hill, NC, USA e Department of Otolaryngology/Head and Neck Surgery, School of Medicine, University of North Carolina at Chapel Hill, 170 Manning Drive, Chapel Hill, NC, USA b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 4 August 2014 Received in revised form 7 October 2014 Accepted 10 October 2014 Available online xxx
Background and objective: Sinus disease is noted to be common in patients with cleft lip and palate. Many have wondered if anatomic differences are a cause or at least a contributor of this. In this sense, comparisons of sinus volumes of patients with different craniofacial clefts may be helpful to determine possible differences from normal. Thus, the present study aimed to evaluate and compare the maxillary sinus volume of patients with unilateral (UCLP) and bilateral (BCLP) cleft lip and palate to control, i.e. non-cleft patients, using cone beam computed tomography (CBCT) images. Methods: The sample consisted of 30 subjects with UCLP, 15 with BCLP and 15 control individuals (noncleft). Each maxillary sinus was assessed three-dimensionally, segmented and its volume was calculated. The comparison between right and left sinus was performed by Student t-test, and the differences between the control and cleft groups were calculated using ANOVA. Results: No statistical differences were found when the sides were compared (p > 0.05). In relation to the assessment among groups, all comparisons had statistically significant differences (p < 0.05), with the UCLP group presenting the lowest sinus volume. Conclusion: UCLP individuals present maxillary sinuses with smaller volumes, without differences found between the cleft and non-cleft side. BCLP subjects also present a reduction in the volume when compared to a control sample, but the average sinus volume is larger than in UCLP patients. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Maxillary sinus Orofacial cleft Cone-beam computed tomography
1. Introduction Patients with cleft lip and palate (CLP) present several anatomical variations in addition to the gap between the nasal and maxillary processes. As an example, alteration in the midface structures is a common finding among affected individuals. These differences may lead to numerous impairments, potentially affecting the health of the ear, nose and throat. Sinus
* Corresponding author. Tel.: +1 919 966 3343. E-mail addresses: [email protected] (G.L. de Rezende Barbosa), [email protected] (L.A. Pimenta), [email protected] (H. Pretti), [email protected] (B.A. Golden), [email protected] (J. Roberts), [email protected] (A.F. Drake).
disease, mainly sinusitis, is frequently noted in CLP patients [1–3]; this highlights the importance of the maxillary sinus, as a major component of the midface and the need of a better understanding of its role in the developmental process and also in the disease. Several studies point to clefting and its role in maxillary sinusitis with evidence indicating many possible etiologic aspects involved. These factors include reflux, sinuses which may be still developing, velopharyngeal insufficiency, presence of pharyngeal flap, maxillary sinus floor height, impaired nasal mucociliary function, as well as hypoplasia of the maxillary sinus [2,4,5]. However, no one factor has been definitively identified as a cause of sinusitis. These mirror the multiple factors that are thought to lead to sinusitis in the non-cleft population.
http://dx.doi.org/10.1016/j.ijporl.2014.10.019 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
G Model
PEDOT-7326; No. of Pages 3 G.L. de Rezende Barbosa et al. / International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
2
As the etiology of sinusitis is not fully understood, and the most common anomaly associated with sinusitis is hypoplasia of the maxillary sinuses [6,7], different ideas have been hypothesized to explain the correlation of sinus disease and the cleft, for example the anatomic differences of the sinuses [1,2]. A significant anatomic variation could demonstrate a potential factor in the higher frequency of sinus disease among patients with cleft. Previous studies evaluated the area of maxillary sinuses of CLP patients using such different imaging modalities as conventional radiographs and multi-slice computed tomography (MSCT) [2,4,5,8–10]. However, the area estimation is a two-dimensional (2D) assessment of a three-dimensional (3D) structure, therefore potentially limited and prone to error. In this sense, a 3D volumetric evaluation of the sinuses is a more accurate option to better understand the complexity of these structures. With the increased presence of cone beam computed tomography (CBCT) in dental practice, more patients with CLP are undergoing CBCT scanning prior to alveolar bone grafting procedures. The CBCT image allows the visualization of structures in all three planes, providing a 3D view of the defect and structures. Different from conventional radiography, which demonstrates a plain image with limited information, CBCT scans are allowing a better appreciation of 3-dimentional anatomy. According to the guidelines of the European Commission in radiation protection of CBCT (SEDENTEXCT), these exams should not be requested for all patients, but for CLP treatment, its use is justified [11]. Moreover, CBCT is also replacing several MSCT studies due to its lower cost and lower radiation dose when compared [12]. The present study aimed to compare the volume of the maxillary sinuses of patients with unilateral (UCLP) and bilateral (BCLP) cleft lip and palate to patients without cleft, using CBCT images, and, in addition, to assess the differences between the right and left sides, and cleft and non-cleft sides. 2. Materials and methods After approval of the study by the Institutional Review Board (IRB#11-1560), 30 subjects with UCLP, 15 with BCLP and 15 control (non-cleft) individuals who underwent CBCT examination comprised the sample of the research. The age ranged from 9 to 12 years old and the groups were age matched. Informed consent regarding the use of CBCT data afterwards was obtained from all patients. Individuals presenting with complete unilateral or bilateral cleft were included in the affected groups, but patients with any diagnosed craniofacial syndrome were excluded from the study. The exclusion criteria for all groups were CBCT exams that did not allow the complete visualization of both maxillary sinuses, presence of mucosal thickening, pseudocysts, retention cysts or any type of sinus pathology, as well as previous surgical interventions in the sinuses and orthodontic expansion treatments. A total
of 75 CBCT exams were observed for selection of the sample and 15 subjects were excluded from the sample (13 UCLP and 2 BCLP) due to complete opacification of the sinus (n = 7), presence of extensive mucosal thickening (n = 5), presence of pseudocyst (n = 1) and insufficient image quality (n = 2); obtaining a final sample of 60 subjects. The CBCT scans of the subjects with cleft were obtained for preoperative planning of reconstructive surgery with alveolar bone grafting, while the exams of the control group were acquired prior routine orthodontic documentation. None of the exam referrals were specifically related to the study. All CBCT images were acquired in a CS9300 unit (Carestream Health, Rochester, NY, USA) operating at 60–90 kVp, 2–15 mA, 0.5 mm slice thickness, 17 cm 13.5 cm field of view and 0.3 voxel size. The obtained scans were loaded onto Mimics software, version 16.0 (Materialize Medical, Leuven, Belgium) and analyzed in the same software. In a secluded room with dim light, a wellcalibrated oral radiologist with experience in tomographic appraisal performed the assessment of the images. The maxillary sinuses were evaluated separately (right/left and cleft/non-cleft), excluding the nasopharyngeal airways and other paranasal sinuses. The threshold was defined manually to include the sinus space and to remove any artifact and background. The volumetric region of interest (VOI) was cropped to comprise only the sinus in question for posterior segmentation. After threshold selection, a three dimensional editing was used to obtain refined surfaces of the segmentation, resulting in a VOI that was rendered into a shaded surface mesh and each segmented volume (cm3) was calculated (Figs. 1 and 2). After one-month interval, 25% of the sample was reassessed to calculate the intra-observer agreement using SigmaStat for Windows (v. 3.5; Systat Software Inc., Erkrath, Germany). All obtained volumetric values were tabulated and Student t-test was performed to compare the volumes between the sides, right and left of control and BCLP groups and cleft and non-cleft sides of UCLP group. The average volume of the sinuses was calculated, resulting in an average value for each patient and ANOVA was used to determine the differences among the volumes of UCLP, BCLP and control (non-cleft) groups. The statistical analyzes were performed using SAS software version 9.4 (Cary, NC, USA). The level of significance was set at p < 0.05. 3. Results The final sample consisted of 60 CBCT images, including 15 control subjects and 45 individuals with orofacial clefts (30 UCLP and 15 BCLP). As a result, a total of 120 sinuses were evaluated. The intra-observer agreement value was 1.0, demonstrating a perfect correlation of the evaluator. The first statistical analysis was performed to compare the volumes of the sinuses between the sides. For this, student t-test was used and no statistical significant differences were found in
Fig. 1. Multiplanar reconstruction of CBCT images demonstrating the contours of the segmented sinuses: sagittal (A), coronal (B) and axial (C) views.
Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
G Model
PEDOT-7326; No. of Pages 3 G.L. de Rezende Barbosa et al. / International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
3
Fig. 2. Coronal (A) and axial (B) views of CBCT images superimposed with the 3D segmentation of the maxillary sinuses and the isolated 3D volume (C). Table 1 LogAverage of the volumes of sinuses and standard error. Group
LogAverage volume (cm3)
Standard error
Control (n = 15) Unilateral (n = 30) Bilateral (n = 15)
4.09 3.79 3.90
0.03 0.02 0.03
any group, neither among right and left sides nor cleft and noncleft sides (p > 0.05). The average volume of the sinuses was then calculated, resulting in one value for each patient. As the distribution of the data was not normal, the log of these values was calculated for statistical evaluation. Next, ANOVA was performed to evaluate the volumes among the three groups: control, UCLP and BCLP. It was observed that all three comparisons had significant differences, where p < 0.0001 when the control group was compared to the UCLP and p = 0.0009 in comparison to the BCLP. In the comparison between the cleft groups (UCLP and BCLP) p = 0.0203. The lowest volume of the sinus was observed in the UCLP group (Table 1).
It was also found that there are no significant differences between the sides, neither between right and left sides in the control and BCLP groups nor cleft and non-cleft sides in the UCLP group. This result differs from Hiosaka et al. findings, who reported larger maxillary sinuses on the non-cleft side in patients with UCLP [10]. On the other hand, our outcomes are in agreement with those of Suzuki et al. and Lee & You, who also observed similar volumes between the sides [5,13]. 5. Conclusion Subjects with CLP, unilaterally and bilaterally, present maxillary sinuses with smaller volumes when compared to age matched control subjects; the UCLP group being the one with smallest dimensions. There are no significant differences between right and left sides and cleft and non-cleft sides found in this study. The authors discuss reasons for variation in these findings in the literature as well as possible causes for the results. As patients with active sinus disease at the time of imaging were excluded from the study, perhaps our findings underestimate the problem in this population.
4. Discussion References The role of sinus development and, ultimately size, in cleft lip and palate patients has been studied previously and authors have proposed possible roles that clefting may play in causing sinusitis [2,4,5]. Authors have proposed contamination of the sinuses by oral secretions, made worse by velopharyngeal insufficiency or an oronasal fistula, as well as impaired mucociliary flow [1,2]. A thorough 3D evaluation of the volume of the maxillary sinuses in this population has not been published. The present study was performed to elucidate the volumetric characteristics of these subjects as well as to compare these findings with previous reports that used different methodologies to assess this anatomic structure. In the current study, significant differences were found between the control group and UCLP and BCLP groups, disagreeing with previous studies that demonstrated no differences in the structure and development of maxillary sinuses between the cleft palate patients and age matched controls [4,13,8]. A possible explanation for the altered volume is the fact that the maxillary sinus develops differently embryonically in cleft palate patients than in those patients with normal palate [12]. Other possible etiologies could be nutritional, as the cleft can cause feeding problems in some patients. A final theoretical possibility could be that this results from altered airflow though the nose and sinuses. Several reports point to the fact that there are no differences in maxillary sinus size between cleft patients and normal populations with both 2D and 3D analyses [2,5]. However, when authors refer to 3D assessments, they are actually considering a 2D measurement (area of the sinus) performed in a 3D exam (MSCT). Conversely, this work aimed to evaluate it differently, performing a real 3D assessment. This difference in the methodology can cause the changes in findings.
[1] B.F. Jaffe, C.B. DeBlanc, Sinusitis in children with cleft lip and palate, Arch. Otolaryngol. 93 (1971) 479–482. [2] Y. Ishikawa, M. Kawano, I. Honjo, R. Amitani, The cause of nasal sinusitis in patients with cleft palate, Arch. Otolaryngol. Head Neck Surg. 115 (1989) 442–446. [3] Y. Ishikawa, R. Amitani, Nasal and paranasal sinus disease in patients with congenital velopharyngeal insufficiency, Arch. Otolaryngol. Head Neck Surg. 120 (1994) 861–865. [4] H. Suzuki, T. Yamaguchi, M. Furukawa, Rhinologic computed tomographic evaluation in patients with cleft lip and palate, Arch. Otolayngol. Head Neck Surg. 125 (1999) 1000–1004. [5] H. Suzuki, T. Yamaguchi, M. Furukawa, Maxillary sinus development and sinusitis in patients with cleft lip and palate, Auris Nasus Larynx 27 (2000) 253–256. [6] H.A. Milczuk, R.W. Dalley, F.W. Wessbacher, M.A. Richardson, Nasal and paranasal sinus anomalies in children with chronic sinusitis, Laryngoscope 103 (1993) 247–252. [7] R.J. Salib, S.A. Chaudri, T.J. Rockley, Sinusitis in the hypoplastic maxillary antrum: the crucial role of radiology in diagnosis and management, J. Laryngol. Otol. 115 (2001) 676–678. [8] H.E. Robinson, G.K. Zerlin, V. Passy, Maxillary sinus development in patients with cleft palates as compared to those with normal palates, Laryngoscope 92 (1982) 183–187. [9] P. Francis, R. Raman, P. Korula, I. Korah, Pneumatization of the paranasal sinuses (maxillary and frontal) in cleft lip and palate, Arch. Otolaryngol. Head Neck Surg. 116 (1990) 920–922. [10] M. Hikosaka, T. Nagasao, H. Ogata, T. Kaneko, K. Kishi, Evaluation of maxillary sinus volume in cleft alveolus patients using 3-dimensional computed tomography, J. Craniofac. Surg. 24 (2013) e23–e26. [11] SEDENTEXCT guidelines. Safety and Efficacy of a New and Emerging Dental X-ray Modality. Radiation protection no. 172: cone beam CT for dental and maxillofacial radiology (evidence-based guidelines), 2012 Available from: http://www. sedentexct.eu/files/radiation_protection_172.pdf. [12] M.A. Kuijpers, A. Pazera, R.J. Admiraal, S.J. Berge´, A. Vissink, P. Pazera, Incidental findings on cone beam computed tomography scans in cleft lip and palate patients, Clin. Oral Invest. 18 (2014) 1237–1244. [13] S.S. Lee, D.S. You, Radiographic study on maxillary sinus development and nasal septum deviation in cleft palate patient, Korean J. Oral Maxillofac. Radiol. 22 (1992) 305–313.
Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
PEDOT-7326; No. of Pages 3 International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Difference in maxillary sinus volumes of patients with cleft lip and palate Gabriella Lopes de Rezende Barbosa a, Luiz Andre´ Pimenta b, Henrique Pretti c, Brent A. Golden d, Jason Roberts e, Amelia Fischer Drake e,* a
Departament of Oral Diagnosis, Division of Oral Radiology, Piracicaba Dental School, University of Campinas, Limeira Avenue, 901, Piracicaba, Sa˜o Paulo, Brazil Department of Dental Ecology, School of Dentistry, University of North Carolina at Chapel Hill, 001 Brauer Hall, Manning Dr & Columbia St. Chapel Hill, NC, USA c Department of Orthodontics, School of Dentistry, Federal University of Minas Gerais, Avenida Antoˆnio Carlos, 6627, Belo Horizonte, Minas Gerais, Brazil d Department of Oral & Maxillofacial Surgery, School of Dentistry, University of North Carolina at Chapel Hill, 149 Brauer Hall, Manning Dr & Columbia St. Chapel Hill, NC, USA e Department of Otolaryngology/Head and Neck Surgery, School of Medicine, University of North Carolina at Chapel Hill, 170 Manning Drive, Chapel Hill, NC, USA b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 4 August 2014 Received in revised form 7 October 2014 Accepted 10 October 2014 Available online xxx
Background and objective: Sinus disease is noted to be common in patients with cleft lip and palate. Many have wondered if anatomic differences are a cause or at least a contributor of this. In this sense, comparisons of sinus volumes of patients with different craniofacial clefts may be helpful to determine possible differences from normal. Thus, the present study aimed to evaluate and compare the maxillary sinus volume of patients with unilateral (UCLP) and bilateral (BCLP) cleft lip and palate to control, i.e. non-cleft patients, using cone beam computed tomography (CBCT) images. Methods: The sample consisted of 30 subjects with UCLP, 15 with BCLP and 15 control individuals (noncleft). Each maxillary sinus was assessed three-dimensionally, segmented and its volume was calculated. The comparison between right and left sinus was performed by Student t-test, and the differences between the control and cleft groups were calculated using ANOVA. Results: No statistical differences were found when the sides were compared (p > 0.05). In relation to the assessment among groups, all comparisons had statistically significant differences (p < 0.05), with the UCLP group presenting the lowest sinus volume. Conclusion: UCLP individuals present maxillary sinuses with smaller volumes, without differences found between the cleft and non-cleft side. BCLP subjects also present a reduction in the volume when compared to a control sample, but the average sinus volume is larger than in UCLP patients. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Maxillary sinus Orofacial cleft Cone-beam computed tomography
1. Introduction Patients with cleft lip and palate (CLP) present several anatomical variations in addition to the gap between the nasal and maxillary processes. As an example, alteration in the midface structures is a common finding among affected individuals. These differences may lead to numerous impairments, potentially affecting the health of the ear, nose and throat. Sinus
* Corresponding author. Tel.: +1 919 966 3343. E-mail addresses: [email protected] (G.L. de Rezende Barbosa), [email protected] (L.A. Pimenta), [email protected] (H. Pretti), [email protected] (B.A. Golden), [email protected] (J. Roberts), [email protected] (A.F. Drake).
disease, mainly sinusitis, is frequently noted in CLP patients [1–3]; this highlights the importance of the maxillary sinus, as a major component of the midface and the need of a better understanding of its role in the developmental process and also in the disease. Several studies point to clefting and its role in maxillary sinusitis with evidence indicating many possible etiologic aspects involved. These factors include reflux, sinuses which may be still developing, velopharyngeal insufficiency, presence of pharyngeal flap, maxillary sinus floor height, impaired nasal mucociliary function, as well as hypoplasia of the maxillary sinus [2,4,5]. However, no one factor has been definitively identified as a cause of sinusitis. These mirror the multiple factors that are thought to lead to sinusitis in the non-cleft population.
http://dx.doi.org/10.1016/j.ijporl.2014.10.019 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
G Model
PEDOT-7326; No. of Pages 3 G.L. de Rezende Barbosa et al. / International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
2
As the etiology of sinusitis is not fully understood, and the most common anomaly associated with sinusitis is hypoplasia of the maxillary sinuses [6,7], different ideas have been hypothesized to explain the correlation of sinus disease and the cleft, for example the anatomic differences of the sinuses [1,2]. A significant anatomic variation could demonstrate a potential factor in the higher frequency of sinus disease among patients with cleft. Previous studies evaluated the area of maxillary sinuses of CLP patients using such different imaging modalities as conventional radiographs and multi-slice computed tomography (MSCT) [2,4,5,8–10]. However, the area estimation is a two-dimensional (2D) assessment of a three-dimensional (3D) structure, therefore potentially limited and prone to error. In this sense, a 3D volumetric evaluation of the sinuses is a more accurate option to better understand the complexity of these structures. With the increased presence of cone beam computed tomography (CBCT) in dental practice, more patients with CLP are undergoing CBCT scanning prior to alveolar bone grafting procedures. The CBCT image allows the visualization of structures in all three planes, providing a 3D view of the defect and structures. Different from conventional radiography, which demonstrates a plain image with limited information, CBCT scans are allowing a better appreciation of 3-dimentional anatomy. According to the guidelines of the European Commission in radiation protection of CBCT (SEDENTEXCT), these exams should not be requested for all patients, but for CLP treatment, its use is justified [11]. Moreover, CBCT is also replacing several MSCT studies due to its lower cost and lower radiation dose when compared [12]. The present study aimed to compare the volume of the maxillary sinuses of patients with unilateral (UCLP) and bilateral (BCLP) cleft lip and palate to patients without cleft, using CBCT images, and, in addition, to assess the differences between the right and left sides, and cleft and non-cleft sides. 2. Materials and methods After approval of the study by the Institutional Review Board (IRB#11-1560), 30 subjects with UCLP, 15 with BCLP and 15 control (non-cleft) individuals who underwent CBCT examination comprised the sample of the research. The age ranged from 9 to 12 years old and the groups were age matched. Informed consent regarding the use of CBCT data afterwards was obtained from all patients. Individuals presenting with complete unilateral or bilateral cleft were included in the affected groups, but patients with any diagnosed craniofacial syndrome were excluded from the study. The exclusion criteria for all groups were CBCT exams that did not allow the complete visualization of both maxillary sinuses, presence of mucosal thickening, pseudocysts, retention cysts or any type of sinus pathology, as well as previous surgical interventions in the sinuses and orthodontic expansion treatments. A total
of 75 CBCT exams were observed for selection of the sample and 15 subjects were excluded from the sample (13 UCLP and 2 BCLP) due to complete opacification of the sinus (n = 7), presence of extensive mucosal thickening (n = 5), presence of pseudocyst (n = 1) and insufficient image quality (n = 2); obtaining a final sample of 60 subjects. The CBCT scans of the subjects with cleft were obtained for preoperative planning of reconstructive surgery with alveolar bone grafting, while the exams of the control group were acquired prior routine orthodontic documentation. None of the exam referrals were specifically related to the study. All CBCT images were acquired in a CS9300 unit (Carestream Health, Rochester, NY, USA) operating at 60–90 kVp, 2–15 mA, 0.5 mm slice thickness, 17 cm 13.5 cm field of view and 0.3 voxel size. The obtained scans were loaded onto Mimics software, version 16.0 (Materialize Medical, Leuven, Belgium) and analyzed in the same software. In a secluded room with dim light, a wellcalibrated oral radiologist with experience in tomographic appraisal performed the assessment of the images. The maxillary sinuses were evaluated separately (right/left and cleft/non-cleft), excluding the nasopharyngeal airways and other paranasal sinuses. The threshold was defined manually to include the sinus space and to remove any artifact and background. The volumetric region of interest (VOI) was cropped to comprise only the sinus in question for posterior segmentation. After threshold selection, a three dimensional editing was used to obtain refined surfaces of the segmentation, resulting in a VOI that was rendered into a shaded surface mesh and each segmented volume (cm3) was calculated (Figs. 1 and 2). After one-month interval, 25% of the sample was reassessed to calculate the intra-observer agreement using SigmaStat for Windows (v. 3.5; Systat Software Inc., Erkrath, Germany). All obtained volumetric values were tabulated and Student t-test was performed to compare the volumes between the sides, right and left of control and BCLP groups and cleft and non-cleft sides of UCLP group. The average volume of the sinuses was calculated, resulting in an average value for each patient and ANOVA was used to determine the differences among the volumes of UCLP, BCLP and control (non-cleft) groups. The statistical analyzes were performed using SAS software version 9.4 (Cary, NC, USA). The level of significance was set at p < 0.05. 3. Results The final sample consisted of 60 CBCT images, including 15 control subjects and 45 individuals with orofacial clefts (30 UCLP and 15 BCLP). As a result, a total of 120 sinuses were evaluated. The intra-observer agreement value was 1.0, demonstrating a perfect correlation of the evaluator. The first statistical analysis was performed to compare the volumes of the sinuses between the sides. For this, student t-test was used and no statistical significant differences were found in
Fig. 1. Multiplanar reconstruction of CBCT images demonstrating the contours of the segmented sinuses: sagittal (A), coronal (B) and axial (C) views.
Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
G Model
PEDOT-7326; No. of Pages 3 G.L. de Rezende Barbosa et al. / International Journal of Pediatric Otorhinolaryngology xxx (2014) xxx–xxx
3
Fig. 2. Coronal (A) and axial (B) views of CBCT images superimposed with the 3D segmentation of the maxillary sinuses and the isolated 3D volume (C). Table 1 LogAverage of the volumes of sinuses and standard error. Group
LogAverage volume (cm3)
Standard error
Control (n = 15) Unilateral (n = 30) Bilateral (n = 15)
4.09 3.79 3.90
0.03 0.02 0.03
any group, neither among right and left sides nor cleft and noncleft sides (p > 0.05). The average volume of the sinuses was then calculated, resulting in one value for each patient. As the distribution of the data was not normal, the log of these values was calculated for statistical evaluation. Next, ANOVA was performed to evaluate the volumes among the three groups: control, UCLP and BCLP. It was observed that all three comparisons had significant differences, where p < 0.0001 when the control group was compared to the UCLP and p = 0.0009 in comparison to the BCLP. In the comparison between the cleft groups (UCLP and BCLP) p = 0.0203. The lowest volume of the sinus was observed in the UCLP group (Table 1).
It was also found that there are no significant differences between the sides, neither between right and left sides in the control and BCLP groups nor cleft and non-cleft sides in the UCLP group. This result differs from Hiosaka et al. findings, who reported larger maxillary sinuses on the non-cleft side in patients with UCLP [10]. On the other hand, our outcomes are in agreement with those of Suzuki et al. and Lee & You, who also observed similar volumes between the sides [5,13]. 5. Conclusion Subjects with CLP, unilaterally and bilaterally, present maxillary sinuses with smaller volumes when compared to age matched control subjects; the UCLP group being the one with smallest dimensions. There are no significant differences between right and left sides and cleft and non-cleft sides found in this study. The authors discuss reasons for variation in these findings in the literature as well as possible causes for the results. As patients with active sinus disease at the time of imaging were excluded from the study, perhaps our findings underestimate the problem in this population.
4. Discussion References The role of sinus development and, ultimately size, in cleft lip and palate patients has been studied previously and authors have proposed possible roles that clefting may play in causing sinusitis [2,4,5]. Authors have proposed contamination of the sinuses by oral secretions, made worse by velopharyngeal insufficiency or an oronasal fistula, as well as impaired mucociliary flow [1,2]. A thorough 3D evaluation of the volume of the maxillary sinuses in this population has not been published. The present study was performed to elucidate the volumetric characteristics of these subjects as well as to compare these findings with previous reports that used different methodologies to assess this anatomic structure. In the current study, significant differences were found between the control group and UCLP and BCLP groups, disagreeing with previous studies that demonstrated no differences in the structure and development of maxillary sinuses between the cleft palate patients and age matched controls [4,13,8]. A possible explanation for the altered volume is the fact that the maxillary sinus develops differently embryonically in cleft palate patients than in those patients with normal palate [12]. Other possible etiologies could be nutritional, as the cleft can cause feeding problems in some patients. A final theoretical possibility could be that this results from altered airflow though the nose and sinuses. Several reports point to the fact that there are no differences in maxillary sinus size between cleft patients and normal populations with both 2D and 3D analyses [2,5]. However, when authors refer to 3D assessments, they are actually considering a 2D measurement (area of the sinus) performed in a 3D exam (MSCT). Conversely, this work aimed to evaluate it differently, performing a real 3D assessment. This difference in the methodology can cause the changes in findings.
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Please cite this article in press as: G.L. de Rezende Barbosa, et al., Difference in maxillary sinus volumes of patients with cleft lip and palate, Int. J. Pediatr. Otorhinolaryngol. (2014), http://dx.doi.org/10.1016/j.ijporl.2014.10.019
