Advertisement

Digital impressions from newborns to preschoolers with cleft lip and palate: A two-centers experience

  • Author Footnotes
    † These authors have contributed equally to this work.
    Benito K. Benitez
    Correspondence
    Corresponding author.
    Footnotes
    † These authors have contributed equally to this work.
    Affiliations
    Oral and Craniomaxillofacial Surgery, University Hospital Basel and University of Basel, Spitalstrasse 21, 4031 Basel, Switzerland

    Department of Clinical Research, University of Basel, Spitalstrasse 12, 4031 Basel, Switzerland

    Department of Biomedical Engineering, University of Basel, Gewerbestrasse 14, 4123 Allschwil, Switzerland
    Search for articles by this author
  • Author Footnotes
    † These authors have contributed equally to this work.
    Andrzej Brudnicki
    Footnotes
    † These authors have contributed equally to this work.
    Affiliations
    Maxillofacial Department, Clinic of Pediatric Surgery, Institute of Mother and Child, ul. Kasprzaka 17a, 01-211 Warsaw, Poland
    Search for articles by this author
  • Zbigniew Surowiec
    Affiliations
    Maxillofacial Department, Clinic of Pediatric Surgery, Institute of Mother and Child, ul. Kasprzaka 17a, 01-211 Warsaw, Poland
    Search for articles by this author
  • Łukasz Wieprzowski
    Affiliations
    Maxillofacial Department, Clinic of Pediatric Surgery, Institute of Mother and Child, ul. Kasprzaka 17a, 01-211 Warsaw, Poland
    Search for articles by this author
  • Abeelan Rasadurai
    Affiliations
    Oral and Craniomaxillofacial Surgery, University Hospital Basel and University of Basel, Spitalstrasse 21, 4031 Basel, Switzerland

    Department of Clinical Research, University of Basel, Spitalstrasse 12, 4031 Basel, Switzerland

    Department of Biomedical Engineering, University of Basel, Gewerbestrasse 14, 4123 Allschwil, Switzerland
    Search for articles by this author
  • Prasad Nalabothu
    Affiliations
    Oral and Craniomaxillofacial Surgery, University Hospital Basel and University of Basel, Spitalstrasse 21, 4031 Basel, Switzerland

    Department of Clinical Research, University of Basel, Spitalstrasse 12, 4031 Basel, Switzerland

    Department of Biomedical Engineering, University of Basel, Gewerbestrasse 14, 4123 Allschwil, Switzerland
    Search for articles by this author
  • Author Footnotes
    † These authors have contributed equally to this work.
    Yoriko Lill
    Footnotes
    † These authors have contributed equally to this work.
    Affiliations
    Oral and Craniomaxillofacial Surgery, University Hospital Basel and University of Basel, Spitalstrasse 21, 4031 Basel, Switzerland

    Department of Clinical Research, University of Basel, Spitalstrasse 12, 4031 Basel, Switzerland

    Department of Biomedical Engineering, University of Basel, Gewerbestrasse 14, 4123 Allschwil, Switzerland
    Search for articles by this author
  • Author Footnotes
    † These authors have contributed equally to this work.
    Andreas A. Mueller
    Footnotes
    † These authors have contributed equally to this work.
    Affiliations
    Oral and Craniomaxillofacial Surgery, University Hospital Basel and University of Basel, Spitalstrasse 21, 4031 Basel, Switzerland

    Department of Clinical Research, University of Basel, Spitalstrasse 12, 4031 Basel, Switzerland

    Department of Biomedical Engineering, University of Basel, Gewerbestrasse 14, 4123 Allschwil, Switzerland
    Search for articles by this author
  • Author Footnotes
    † These authors have contributed equally to this work.
Open AccessPublished:August 21, 2022DOI:https://doi.org/10.1016/j.bjps.2022.08.015

      Summary

      Background

      Documenting cleft lip and palate morphology prior to surgery is standard care. Presurgical orthopedic treatment also requires a 3D cleft model. Endangering the airway, conventional impressions require additional safety measures and resources. We investigate the implementation and risks of digital impressions for the youngest patients with orofacial clefts.

      Methods

      We report a retrospective cohort study of patients with cleft lip and palate, aged up to 6 years, treated at two cleft centers in Europe (Basel (A), Warsaw (B)). We scanned with the Medit i500 (Medit Corp, Seoul, South Korea). Center A for presurgical orthopedics and prior surgery from June 2020 to March 2022. Center B prior surgery from December 2020 to May 2021. Scanning data were analyzed for adverse events and adverse device effects, scanning duration, and number of images according to cleft type and age.

      Results

      We analyzed 342 digital impressions in 190 patients (center A: 71, B: 119). The median age was 8.7 months with a range from the first day of birth (presurgical orthopedics) to six years of life (early alveolar bone grafting). No adverse events or adverse device effects were observed. The median scan duration was 85.5 s for cleft palate and 50 s for cleft lip and nose (IQR 56 s and 39 s, respectively).

      Conclusion

      Digital impressions with intraoral scanners are safe in patients with cleft lip and palate from newborn to preschool age. Given the funding to purchase an intraoral scanner, interfaces to electronic patient records, and point-of-care 3D printing, cleft centers can successfully implement this technology.

      Keywords

      Introduction

      Documenting morphological changes in cleft lip and palate due to growth and interventions allows assessment of treatment quality and outcome research. The World Health Organization (WHO) has established recommendations for documentation along with timings in cleft care protocols. In addition to records, such as radiology, speech therapy, and audiology, these include 2D photos and 3D models.
      World Health Organization
      Global strategies to reduce the health-care burden of craniofacial anomalies: report of WHO meetings on International Collaborative Research on Craniofacial Anomalies, Geneva, Switzerland, 5-8 November 2000; Park City, utah, U.S.A., 24-26 May 2001.
      The Cleft Steering Group of the Royal College of Surgeons and the Craniofacial Society of Great Britain and Ireland also recommended making a model of the cleft deformity prior surgery.
      • Staiano J.J.
      • Gravenor C.
      • Milling M.A.P.
      Standardisation of cleft care-a technique for creating a three-dimensional model of the cleft lip, palate and nose.
      Therefore, conventional impressions for plaster cast models have been used for years, which enabled intercenter comparisons.
      • Daskalogiannakis J.
      • Mercado A.
      • Russell K.
      • et al.
      The americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 3. Analysis of craniofacial form.
      • Shaw W.C.
      • Semb G.
      • Nelson P.
      • et al.
      The Eurocleft project 1996-2000: overview.
      • Fudalej P.S.
      • Urbanova W.
      • Klimova I.
      • et al.
      The Slavcleft: a three-center study of the outcome of treatment of cleft lip and palate. Part 2: dental arch relationships.
      Presurgical orthopedic treatment, such as nasoalveolar molding, further requires repeated impressions.
      • Naveau A.
      • Grémare A.
      • Plaire V.
      • Ducret M.
      • Loot Maya
      • Noirrit-Esclassan E
      Digital management of low cost presurgical plates for young patients with palatal cleft.
      Digital impressions with intraoral scanners have been replacing conventional impression methods in dentistry and orthodontics since their introduction in the 1980s.
      • Birnbaum N.S.
      • Aaronson H.B.
      Dental impressions using 3D digital scanners: virtual becomes reality.
      Although the use of intraoral scanners has become common in dentistry, their implementation has been lagging for patient care in orofacial malformations due to technical hurdle in the reconstruction of intraoral structure without teeth. Only in recent years, their use for infants with cleft lip and palate has been explored increasingly.
      • Yilmaz H.
      • Aydin M.N.
      Digital versus conventional impression method in children: comfort, preference and time.
      • Xepapadeas A.B.
      • Xepapadeas A.B.
      • Weise C.
      • et al.
      Technical note on introducing a digital workflow for newborns with craniofacial anomalies based on intraoral scans - Part II: 3D printed Tübingen palatal plate prototype for newborns with Robin sequence.
      • Patel J.
      • Winters J.
      • Walters M.
      Intraoral digital impression technique for a neonate with bilateral cleft lip and palate.

      Krey K.-.F., Ratzmann A., Metelmann P.H., Hartmann M., Ruge S., Kordaß B. Fully digital workflow for presurgical orthodontic plate in cleft lip and palate patients. Int J Comput Dent. 21(3):251–259.

      • Chalmers E.V.
      • McIntyre G.T.
      • Wang W.
      • Gillgrass T.
      • Martin C.B.
      • Mossey P.A.
      Intraoral 3D scanning or dental impressions for the assessment of dental arch relationships in cleft care: which is superior?.

      Shanbhag G., Pandey S., Mehta N., Kini Y., Kini A. A virtual noninvasive way of constructing a nasoalveolar molding plate for cleft babies, using intraoral scanners, CAD, and prosthetic milling. Cleft Palate-Craniofacial J. Published online 2019. doi:10.1177/1055665619886476.

      • Choi Y.S.
      • Shin H.S.
      Preoperative planning and simulation in patients with cleft palate using intraoral three-dimensional scanning and printing.
      Digital impression using intraoral scanners allows for the contactless acquisition of intraoral as well as lip and nose morphology of the patients as opposed to the traditional technique by physical impressions which accompanies a risk of endangering the airway of the infants with impression material.
      • Radojicic J.
      Cleft Care: intraoral 3D scanning.
      • Reichert F.
      • Amrhein P.
      • Uhlemann F.
      Unnoticed aspiration of palate plate impression material in a neonate: diagnosis, therapy, outcome.
      • Schober P.
      • Vetter T.R.
      Survival analysis and interpretation of time-to-event data: the tortoise and the hare.
      • Chate R.A.
      A report on the hazards encountered when taking neonatal cleft palate impressions (1983-1992).
      The ease of operating the scanner mitigates the conventional, elaborate procedure involving a multidisciplinary team on-site, allowing for more frequent patient data acquisition. Considering the availability of various imaging modalities and the digitalization of patient records in general, digital impression of intraoral morphology is a natural replacement for the conventional impression taking for plaster casts.
      Chalmers et al. have evaluated the use of an intraoral scanner for patients with cleft lip and palate at the age between 5 and 21 years in comparison with conventional impressions.
      • Chalmers E.V.
      • McIntyre G.T.
      • Wang W.
      • Gillgrass T.
      • Martin C.B.
      • Mossey P.A.
      Intraoral 3D scanning or dental impressions for the assessment of dental arch relationships in cleft care: which is superior?.
      Since then, the intraoral scanner has been gaining acceptance for use in infants for the treatment of cleft lip and palate. There have been several reports on single to few cases in infants with cleft lip and palate in the context of developing digital workflows for the fabrication of orthopedic appliances.
      • Patel J.
      • Winters J.
      • Walters M.
      Intraoral digital impression technique for a neonate with bilateral cleft lip and palate.
      ,
      • Dalessandri D.
      • Tonni I.
      • Laffranchi L.
      • et al.
      Evaluation of a digital protocol for pre-surgical orthopedic treatment of cleft lip and palate in newborn patients: a pilot study.
      ,
      • Gong X.
      • Dang R.
      • Xu T.
      • Yu Q.
      • Zheng J.
      Full digital workflow of nasoalveolar molding treatment in infants with cleft lip and palate.
      Recently, Weise et al. have reported an evaluation of intraoral scanning in infant to small children with craniofacial malformations.
      • Weise C.
      • Frank K.
      • Wiechers C.
      • et al.
      Intraoral scanning of neonates and infants with craniofacial disorders: feasibility, scanning duration, and clinical experience.
      However, the assessment of the integration of digital impression taken with intraoral scanners in clinical routines is still scarce, thereby limiting the feasibility reported to a handful of cases.
      We report the implementation of digital impression taking with intraoral scanners in surgeons’ hands in routine clinical workflow in a medium and a large cleft center. We investigated the safety and scanning time in awake patients and under anesthesia from newborns to preschoolers with cleft lip and palate. We assessed whether digital impressions could capture high-quality lip/nose and intraoral cleft morphology that can be used for documentation, designing orthopedic appliances, and monitoring treatment effects.

      Patients and methods

      Study design, setting, and participants

      We report according to the STROBE guidelines for cohort studies.
      • von Elm E.
      • Altman D.G.
      • Egger M.
      • Pocock S.J.
      • Gøtzsche P.C.
      • Vandenbroucke J.P.
      The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies.
      In this retrospective cohort study, we assessed the implementation of an intraoral scanner to digitally record cleft malformations at two cleft centers (centers A and B). At center A, digital impressions were implemented in June 2020, and data collected until March 2022 are included in this report. Scanning has been routinely performed in patients with orofacial clefts for presurgical orthopedics and at the operation theater under anesthesia before primary cleft repair and early alveolar bone grafting. At center B, scans were taken at the operation theater under anesthesia before primary cleft repair and alveolar bone grafting. Data collected from December 2020 to May 2021 at center B are included in the report. Scanning was performed by cleft surgeons (maxillofacial surgeons and pediatric surgeons) who were present at either the consultation or at the surgery. Inclusion criteria were scanning of cleft malformation with an intraoral scanner in neonates and children up to six years of age with a signed informed-consent form of parents for use of the data in scientific investigations. The study was performed in accordance with the Declaration of Helsinki after obtaining approval from the local and institutional ethics committee (Ethics Committee of Northwest and Central Switzerland project: AO_2020 and 2021–01,988 and The Bioethics Committee of the Institute of Mother and Child (IMC), reference 47/2020).

      Variables

      Baseline characteristics of the patients, cleft type, diagnosis of Robin sequence, reason for scan, physician performing the scan, scanning time, numbers of images/frames required for 3D rendering per scan, and adverse events as well as adverse device effects (according to EN ISO 14,155:2020

      ISO. Clinical investigation of medical devices for human subjects - Good clinical practice. Bs En Iso 14155-2:2009. Published 2011. Accessed March 17, 2022. https://www.iso.org/standard/71690.html

      ) were recorded in the electronic Case Report Form (eCRF). The data were collected and managed using Research Electronic Data Capture (REDCap®) tools.
      • Harris P.A.
      • Taylor R.
      • Thielke R.
      • Payne J.
      • Gonzalez N.
      • Conde J.G.
      Research electronic data capture (REDCap)-A metadata-driven methodology and workflow process for providing translational research informatics support.
      ,
      • Harris P.A.
      • Taylor R.
      • Minor B.L.
      • et al.
      The REDCap consortium: building an international community of software platform partners.

      Data sources and measurements

      Digital impressions of the 3D cleft morphology of newborns to preschoolers were acquired with an intraoral scanner (Medit i500, Medit Corp, Seoul, South Korea). At center A, scanning was performed in cases of presurgical orthopedic treatment on the day or a few days after birth and at around 4 months of age for plate renewal.
      • Nalabothu P.
      • Benitez B.K.
      • Dalstra M.
      • Verna C.
      • Mueller A.A.
      Three-dimensional morphological changes of the true cleft under passive presurgical orthopaedics in unilateral cleft lip and palate: a retrospective cohort study.
      At both centers A and B, scanning was performed in the operating theater prior to primary cleft repair and early alveolar bone grafting. Comparability of both groups is given as both centers performed single-staged cleft lip and palate repair at around 8 months of age and early alveolar bone grafting before the age of 7 years.
      • Benitez B.K.
      • Brudnicki A.
      • Surowiec Z.
      • et al.
      Continuous circular closure in unilateral cleft lip and plate repair in one surgery.
      ,
      • Brudnicki A.
      • Regulski P.A.
      • Sawicka E.
      • Fudalej P.S.
      Alveolar volume following different timings of secondary bone grafting in patients with unilateral cleft lip and palate. A pilot study.
      Figure 1 and Supplemental Digital Content show examples of digital impressions of nose and lip (Video 1) as well as intraoral scan of cleft palate (Video 2) with merged nose-lip scan with the palate in a patient with unilateral cleft lip and palate (Video 3) and bilateral cleft lip and palate (Video 4).
      Fig 1:
      Fig. 1Digital impression examples of nose and lip as well as cleft palate scan of infants with (A, B) unilateral cleft lip and palate (B, C) bilateral cleft lip and palate scan (C, D) cleft palate.

      Quantitative variables

      The scanning duration for each scan was extracted from the scan records. It included the time the scanner head was turned on to capture images to render 3D morphology. We further extracted the number of images/frames captured during the scanning for each 3D rendering. Subgroup analysis was performed to test whether the scanning duration and number of frames required for 3D rendering in each scan depended on whether the patients were under anesthesia during the scans, as well as at which center the scanning was performed. Further, we analyzed whether scan duration depended on cleft type or the age of the patients. For comparability with previously reported studies, we divided the patients into three age groups as described in Weise et al.
      • Weise C.
      • Frank K.
      • Wiechers C.
      • et al.
      Intraoral scanning of neonates and infants with craniofacial disorders: feasibility, scanning duration, and clinical experience.
      with an addition of a preschool age group; (1) birth up to 28 days, (2) 29–365 days, (3) 1–3 years old, and (4) 3–6 years old. We categorized the patients and scans according to cleft types to study their effect on scanning procedure: (1) soft palate cleft, (2) soft and hard palate cleft, (3) unilateral cleft lip and palate, (4) bilateral cleft lip and palate, and (5) cleft lip and alveolus.

      Statistical methods

      The primary outcome, implementation of intraoral scanner used in routine consultation and operation theater, is measured by safety (adverse events and adverse device effects) and scanning time with number of frames per scan and is analyzed using descriptive statistics. Wilcoxon rank-sum test and Kruskal–Wallis test were conducted for scanning duration and number of images/frames per scan, whether they depend on the center, whether the patients were awake or under anesthesia, cleft type, and the age of the patients.

      Results

      Participants and descriptive data

      We analyzed a total of 342 digital impressions in 190 patients. There were 71 patients with 146 scans from the medical records of center A and 119 patients with 196 scans from center B. Table 1 shows the patients characteristics. A graphical representation of number of patients and number of scans for each cleft type, with the distribution of scans depending on age, are shown in the supplementary material (Figure S 1).
      Table 1Baseline characteristics of the patients scanned at center A and center B.
      CharacteristicTotalCenter ACenter B
      Number of patients19071119
      % Male595960
      % Female414140
      Cleft types (number of patients):
      Soft palate cleft17512
      Soft and hard palate cleft361620
      Unilateral cleft lip and palate632439
      Bilateral cleft lip and palate401030
      Cleft lip and alveolus19811
      Cleft lip1587
      Table 2 summarizes the details of the scans comparing the two centers. The median age of the patients at scanning was 9 months (interquartile range IQR 27.14 months). No adverse device events or adverse events were recorded, and no scan had to be repeated. Table 2 further depicts the purposes of the scanning with 37 scans performed at birth or at a few weeks old for designing presurgical orthopedic appliances, including 5 patients with Robin sequence which were for pre-epiglottic baton plate therapy.

      Poets C.F., Wiechers C., Koos B., Muzaffar A.R., Gozal D. Pierre Robin and breathing: what to do and when? Pediatr Pulmonol. Published online 2021. doi:10.1002/ppul.25317.

      A total of 118 scans preceded a surgical cleft repair and 41 scans preceded alveolar bone grafting. Reasons for scanning under other included scans for post-alveolar bone grafting control, early cleft rhinoplasty, and secondary cleft corrections.
      Table 2Comparison between centers, age at scan, scan numbers, and durations.
      TotalCenter ACenter B
      Age at scan in months; median(IQR)8.74(27.14)8.06(54.29)9.13(25.89)
      Total number of digital impressions342146196
      Number of palate scans21899119
      Number of lip and nose scans1244777
      Scan duration in sec.; median(IQR)
      Palate85.5(56)115(58)70(35.5)
      Lip and nose50(39)78(68)44(27)
      Number of frames/scan; median(IQR)
      Palate1681 (1114.5)2143(1444)1420.5(708.25)
      Lip and nose1199(772.5)1522(1180)1029(617.75)
      Reasons for scanning (palate):
      Presurgical orthopedics37370
      Primary cleft repair1183286
      Alveolar bone grafting411724
      Other21138
      Number of palate scans in:
      Awake patients50500
      Under anesthesia16849119
      Adverse events/adverse device effects0/00/00/0

      Main results: safety and influence on scan duration

      There were no adverse events or adverse device effects recorded during scanning at both centers. Digital impressions have completely replaced conventional impression taking in center A after the first three digital impressions for newborns were taken. Thereafter, no patient required conventional impressions. The median scan duration was 85.5 s for cleft palate scan and 50 s for cleft lip and nose (IQR 56 s and 39 s, respectively). Systematic scanning of the complete maxillary arch from one tuberosity to the other, including the entire cleft palate surface, could be obtained within 2 min. Figure 2 shows the scan duration needed for capturing cleft palate morphology depending on whether the patients were awake or under anesthesia. The results of statistical analyses are summarized in Table S 1. Wilcoxon rank-sum test on the subgroup indicates that there are statistically significant differences in the median time taken for each palate scan and for each nose/lip scan at center A compared to center B (p-value < 0.05). Visual inspection based on boxplots indicates that there is no difference in the dependency of scan duration on age group or cleft type between the centers (Figure S 2). The data from center A and Wilcoxon rank sum test show that scan duration does not statistically significantly differ whether the scans were acquired while the patients were awake or under anesthesia (Figure S 2 (B), p-value > 0.05).
      Fig 2:
      Fig. 2Boxplot comparing scan duration between center A and center B depending on whether the patients were awake or under anesthesia (A) cleft lip and nose scan (B) cleft palate scan.
      Figure 3 and Table 3 show scan duration depending on the age group and cleft type in both centers.
      Fig 3:
      Fig. 3Boxplots showing the distribution of scanning time of lip and nose depending on (A) age (B) cleft type as well as intraoral scanning time depending on (C) age and (D) cleft type of the patients in both centers.
      Table 3Scan durations and number of frames per scan depending on the age group and cleft type.
      Neonates (0–28 days)Infants (29–365 days)Small children (1–3 years)Preschoolers (4–6 years)
      Number of scans:
       Palate181263935
       Lip & nose scans9791719
      Scan duration in sec.; median(IQR):
       Palate94(52)75.5(53)79(34.5)124(70)
       Lip and nose12(18)50(36.5)48(33)95(59)
      Number of frames/scan; median(IQR):
       Palate1772.5(1154)1448(937.75)1724(606.5)2577(1863.5)
       Lip and nose171(304)1183(734.25)1113(734)1854(1041)
      Soft palate cleftSoft & hard palate cleftUnilateral cleft lip & palateBilateral cleft lip & palateCleft lip & alveolusCleft lip
      Number of scans:
       Palate175274421914
       Lip & nose scans31252301611
      Scan duration in sec.; median(IQR):
       Palate70(42)73.5(55)92(56)89(62.5)70(75.5)67.5(61.5)
       Lip and nose74(16.5)46(48.5)54.5(54)49.5(20.25)53.5(40.25)47(72.5)
      Number of frames/scan; median(IQR):
       Palate1580(552)1439(932.5)1809(1125)1826(1183)1721(1489.5)1315.5(1426.5)
       Lip and nose1445(279)1185(901)1210(821)1243(636)1155.5(872.25)1046(1251)
      Visual inspection of boxplots of the time required for scanning lip and nose shows that the younger the patient, the shorter the scan duration, since the area to be scanned is smaller for younger patients. Cleft type does not affect the time it takes for scanning lip and nose. On the other hand, intraoral scanning becomes more difficult in younger patients with smaller intraoral space to maneuver the scanner head; consequently, a longer scan duration is needed.
      Subgroup analyses with Kruskal–Wallis test by ranks were conducted to examine the effect of age and cleft type on the time needed for scanning the palate. Dependency of the scan duration on age and cleft type did not differ between the two centers based on visual inspection by the boxplots (Figure S 2). We performed the tests separately for subgroups to control for the effect of difference in distribution of scan numbers between the two centers. Therefore, the subgroups analyzed were scans in (1) awake patients in center A, (2) patient under anesthesia in center A, and (3) patients under anesthesia in center B. Among scans performed in center A on awake patients and patients under anesthesia, there was no difference among age groups and among cleft types (p-value > 0.05), except for awake patients among cleft types (p-value = 0.0037). Among the scans performed in center B, which were all under anesthesia, we found that age group has no effect on the scan duration (p-value > 0.05), but cleft type does (p-value = 0.0016).

      Discussion

      We implemented digital impression taking with intraoral scanners in surgeons’ hands at a medium and large interdisciplinary cleft center. We assessed whether digital impressions can capture high-quality three-dimensional cleft morphology for patient records and to design orthopedic appliances. We investigated safety and scan time in newborns to preschoolers with cleft lip and palate, both awake and under anesthesia.
      Digital impressions have completely replaced conventional impression taking in center A after the first three digital impressions for newborns were taken. High-quality 3D intraoral scans could be captured without adverse events in newborns and infants while they are awake, on average within 100 s. Scanning of lip and nose takes an additional 50 s. Acquisition of nose/lip and palate malformation allowed for low-risk data acquisition without soft tissue distortion. The colored digital dataset could be archived with a high accuracy. Further integration into a digital workflow also gave the possibility to digitally design and 3D print presurgical orthopedic appliances[
      • Zarean Parichehr
      • Zarean Paridokht
      • Thieringer Florian M.
      • Mueller Andreas A.
      • Kressmann Sabine
      • Erismann Martin
      • Sharma Neha
      • Benitez Benito K.
      A Point-of-Care Digital Workflow for 3D Printed Passive Presurgical Orthopedic Plates in Cleft Care.
      ].
      In centers A and B, digital impression taking was implemented in the operating theater for recording before cleft surgery. No adverse events occurred and scanning duration under anesthesia for capturing intraoral morphology was as short as 70 s on average. Scanning duration can vary depending on the age due to the relative size of the scanner head and patients’ mouth affecting the efficiency to capture the images for 3D reconstruction. The 3D reconstruction takes place while capturing images of the object within the depth of field of the sensor. The reconstruction pauses whenever the object is further away than the maximum distance until the object re-enters of the depth of field. The average framerate for a scan depends on the amount of time spent without collecting images contributing to the 3D reconstruction and, hence, indicates the efficiency of scanning. If the patients are under anesthesia, the framerate did not differ between the centers (Figure S2 (A), p-value > 0.05), which indicates that the efficiency to reconstruct 3D morphology during scanning did not depend on the surgeon guiding the scanner. Comparison between the framerate in awake patient vs. patients under anesthesia indicates that the ease of scanning under anesthesia may contribute to less time loss due to scanner head moving out of the depth of field (Figure S2 (B), p-value < 0.05). The simultaneous visual feedback of the scanner head position relative to the surface being scanned can assist in increasing the average framerate and, hence, improve the efficiency.

      Clinical relevance for cleft lip and palate repair

      Patients with orofacial malformation and their families are confronted with series of treatment steps. Any advancement to reduce burden and risk and enhance treatment outcomes is exceptionally valuable, especially at their early ages. Thus, alternatives to conventional impressions are in need,
      • Radojicic J.
      Cleft Care: intraoral 3D scanning.
      since they are associated with known complications, require technical know-how (impression material, individual trays, and dental training) and resources with readiness for airway management. Due to these circumstances, conventional impressions are no longer practiced in certain centers.
      • Reichert F.
      • Amrhein P.
      • Uhlemann F.
      Unnoticed aspiration of palate plate impression material in a neonate: diagnosis, therapy, outcome.
      The simplified digital impression not only benefits the patients but also digitalization allows leveraging of technological advancements.
      • Xepapadeas A.B.
      • Xepapadeas A.B.
      • Weise C.
      • et al.
      Technical note on introducing a digital workflow for newborns with craniofacial anomalies based on intraoral scans - Part II: 3D printed Tübingen palatal plate prototype for newborns with Robin sequence.
      ,
      • Gong X.
      • Dang R.
      • Xu T.
      • Yu Q.
      • Zheng J.
      Full digital workflow of nasoalveolar molding treatment in infants with cleft lip and palate.
      ,
      • Shen C.
      • Yao C.A.
      • Magee W.
      • Chai G.
      • Zhang Y.
      Presurgical nasoalveolar molding for cleft lip and palate: the application of digitally designed molds.
      • Ayoub A.F.
      • Garrahy A.
      • Hood C.
      • et al.
      Validation of a vision-based, three-dimensional facial imaging system.
      • Ayoub A.
      • Khan A.
      • Aldhanhani A.
      • et al.
      The validation of an innovative method for 3D capture and analysis of the nasolabial region in cleft cases.
      3D imaging provides new and enhanced outcome analysis of the facial aesthetics of children with cleft.
      • Sharma V.P.
      • Bella H.
      • Cadier M.M.
      • Pigott R.W.
      • Goodacre T.E.E.
      • Richard B.M.
      Outcomes in facial aesthetics in cleft lip and palate surgery: a systematic review.
      The possibility of combining a digital impression of the face with the palate prior to surgery allows to obtain a more complete record of the combined cleft lip and palate deformity.
      • Staiano J.J.
      • Gravenor C.
      • Milling M.A.P.
      Standardisation of cleft care-a technique for creating a three-dimensional model of the cleft lip, palate and nose.
      As a further advantage, both the anatomy and the color information are integrated into the digital 3D model. Nevertheless, the evaluation of this new technology with regard to the benefit and implication for the young patients, in addition to logistical issues in various types of centers, is a prerequisite for successful clinical transfer. If the disadvantages of procurement costs and implementation into the clinical workflow can be overcome, this technology has the clear potential to further improve treatment.

      Limitations

      Although the data were collected from two centers, the number of scans in the dataset was too small to reach reliable statistical result for studying the effect of age and cleft type on the scanning performance measured by scanning duration and number of frames required for each 3D reconstruction. With further implementation and results from other centers, we should be able to test our findings for consistency and allow for more reliable analysis of scanning procedures. Our results are specific to Medit i500; therefore, the implementation and the adequacy of a scanner for patients with orofacial malformation at their young ages may differ depending on the model. Issues involved in implementation and its feasibility for these patients will depend on the choice of the model also within the same producer. Nevertheless, with newer models, a faster acquisition and smaller scan tips can be expected to be even better suited for the youngest patients with clefts.

      Conclusion

      As a cleft care provider, the message is clear: intraoral scan technology has clear advantages over conventional impression taking. If the hurdles of acquisition costs and interfaces in the workflow can be overcome, conventional impressions will be replaced in clinical routine. We successfully implemented digital impression taking with an intraoral scanner in a medium and large cleft center by surgeons in the operation theater and routine consultations. An established workflow for point-of-care 3D printing was a prerequisite for ensuring therapy with presurgical orthopedics. The choice of a particular intraoral scanner depends on needs specific for these newborns and infants as well as the feasibility of implementation in clinical routine. These include but are not limited to the size of the scanner head, framerate to speed up capturing procedure, imaging depth to reconstruct the complete morphology, data handling, software capabilities, and file formats. In conclusion, the benefit of digital impressions is multi-faceted:
      • Airway safe use for digital image acquisition from newborns to preschoolers with clefts,
      • Simplifies the procedure and saves time due to direct digitally storable data acquisition,
      • Eliminates the involvement of multi-disciplinary personnel for acquiring 3D morphology,
      • Scanning can be performed in an operation theater by surgeons to evaluate their outcome,
      • Produce high-quality digital images with additional color information,
      • Scanning can combine and merge lip and nose with the cleft palate,
      • Digital workflow integration with point-of-care 3D printing and electronic medical records.
      Digital impressions obtained by scanners can be employed in treating children with cleft lip and palate for diagnostic purpose, treatment planning, anatomical analysis, and fabrication of presurgical orthopedic appliances. In addition to the practical aspect, digital documentation enables healthcare providers to leverage the advancement in digital technologies at the forefront to further improve treatment strategies.

      CRediT authorship contribution statement

      Benito K. Benitez: Conceptualization, Methodology, Validation, Formal analysis, Investigation, Data curation, Visualization, Project administration, Funding acquisition, Writing – original draft. Andrzej Brudnicki: Supervision, Conceptualization, Investigation, Methodology, Resources, Writing – review & editing. Zbigniew Surowiec: Investigation, Resources, Writing – review & editing. Łukasz Wieprzowski: Validation, Investigation, Data curation, Writing – review & editing. Abeelan Rasadurai: Formal analysis, Data curation, Writing – review & editing. Prasad Nalabothu: Writing – review & editing. Yoriko Lill: Validation, Formal analysis, Data curation, Visualization, Writing – original draft. Andreas A. Mueller: Supervision, Conceptualization, Methodology, Investigation, Resources, Funding acquisition, Writing – review & editing.

      Declaration of competing interest

      The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as potential conflicts of interest.

      Funding

      This study was supported by the Botnar Research Centre for Child Health (BRCCH), supporting the Research Consortium for pediatric digital health with a Multi-Investigator Project, “Burden-Reduced Cleft Lip and Palate Care and Healing” led by Andreas A. Mueller. Further support was obtained from the Propatient Research Foundation of the University Hospital Basel, where Benito K. Benitez acknowledges that he received financial support for the clinical implementation of contactless impression taking and 3D printing for cleft lip and palate malformations by the Propatient Research Foundation of the University Hospital Basel (pp 19–11 Contactless Impressions and 3D Printing in Cleft Lip and Palate). Benito K. Benitez acknowledges that he received financial support for his PhD program through a grant from the Department of Surgery at the University Hospital of Basel. The funding sources were not involved in the study design, the collection, analysis, or interpretation of data, the preparation of the manuscript, or the decision to submit the manuscript for publication. This research did not receive any other specific grants from funding bodies in the public, commercial, or non-profit sectors.

      Ethical Approval

      The study was performed in accordance with the Declaration of Helsinki after obtaining approval from the local and institutional ethics committee (Ethics Committee of Northwest and Central Switzerland project: AO_2020 and 2021–01988 and The Bioethics Committee of the Institute of Mother and Child (IMC), reference 47/2020.

      Acknowledgements

      The authors thank the Pediatric Surgery team of the Institute of Mother and Child, Warsaw, Poland, for their support: Professor Ewa Sawicka MD PhD, Włodzimierz Piwowar MD PhD, Barbara Offert MD, and Orest Szczygielski MD PhD. We further thank Sabine Kressmann PhD for the project management for successful clinical implementation of the technology in daily routine at the University Hospital Basel and University Children's Hospital Basel. The authors thank the Propatient Research Foundation of the University Hospital Basel and Botnar Research Centre for Child Health for their support.

      Appendix. Supplementary materials

      • Figure, Supplementary Digital Content 1: Number of (A) patients and (B) scans included in the study according to cleft type (C) scans included in the study according to age at scanning.

      • Figure, Supplementary Digital Content 2: Effect of covariates shown by boxplots for scan durations of palates; dependency on center for patients under anesthesia with factor being (A) cleft type and (B) age group, dependency on whether the patients were awake or under anesthesia in center A with factor being (C) cleft type (D) age group.

      • Figure, Supplementary Digital Content 3: Boxplots to compare average framerate per scan for lip and nose (A) and palate scan (B) achieved at center A vs. center B and while patients are awake vs. under anesthesia.

      References

        • World Health Organization
        Global strategies to reduce the health-care burden of craniofacial anomalies: report of WHO meetings on International Collaborative Research on Craniofacial Anomalies, Geneva, Switzerland, 5-8 November 2000; Park City, utah, U.S.A., 24-26 May 2001.
        World Health. 2002; : 1-148
        • Staiano J.J.
        • Gravenor C.
        • Milling M.A.P.
        Standardisation of cleft care-a technique for creating a three-dimensional model of the cleft lip, palate and nose.
        J Plast Reconstr Aesthetic Surg. 2006; 59: 826-828https://doi.org/10.1016/j.bjps.2005.11.011
        • Daskalogiannakis J.
        • Mercado A.
        • Russell K.
        • et al.
        The americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 3. Analysis of craniofacial form.
        Cleft Palate-Craniofacial J. 2011; 48: 252-258https://doi.org/10.1597/09-185.1
        • Shaw W.C.
        • Semb G.
        • Nelson P.
        • et al.
        The Eurocleft project 1996-2000: overview.
        J Cranio-Maxillofacial Surg. 2001; 29: 131-140https://doi.org/10.1054/jcms.2001.0217
        • Fudalej P.S.
        • Urbanova W.
        • Klimova I.
        • et al.
        The Slavcleft: a three-center study of the outcome of treatment of cleft lip and palate. Part 2: dental arch relationships.
        J Cranio-Maxillofacial Surg. 2019; 47: 1092-1095https://doi.org/10.1016/j.jcms.2019.03.023
        • Naveau A.
        • Grémare A.
        • Plaire V.
        • Ducret M.
        • Loot Maya
        • Noirrit-Esclassan E
        Digital management of low cost presurgical plates for young patients with palatal cleft.
        French J Dent Med. 2021; : 1-6https://doi.org/10.36161/FJDM.0008
        • Birnbaum N.S.
        • Aaronson H.B.
        Dental impressions using 3D digital scanners: virtual becomes reality.
        Compend Contin Educ Dent. 2008; 29
        • Yilmaz H.
        • Aydin M.N.
        Digital versus conventional impression method in children: comfort, preference and time.
        Int J Paediatr Dent. 2019; 29: 728-735https://doi.org/10.1111/ipd.12566
        • Xepapadeas A.B.
        • Xepapadeas A.B.
        • Weise C.
        • et al.
        Technical note on introducing a digital workflow for newborns with craniofacial anomalies based on intraoral scans - Part II: 3D printed Tübingen palatal plate prototype for newborns with Robin sequence.
        BMC Oral Health. 2020; 20https://doi.org/10.1186/s12903-020-01159-7
        • Patel J.
        • Winters J.
        • Walters M.
        Intraoral digital impression technique for a neonate with bilateral cleft lip and palate.
        Cleft Palate-Craniofacial J. 2019; 56: 1120-1123https://doi.org/10.1177/1055665619835082
      1. Krey K.-.F., Ratzmann A., Metelmann P.H., Hartmann M., Ruge S., Kordaß B. Fully digital workflow for presurgical orthodontic plate in cleft lip and palate patients. Int J Comput Dent. 21(3):251–259.

        • Chalmers E.V.
        • McIntyre G.T.
        • Wang W.
        • Gillgrass T.
        • Martin C.B.
        • Mossey P.A.
        Intraoral 3D scanning or dental impressions for the assessment of dental arch relationships in cleft care: which is superior?.
        Cleft Palate-Craniofacial J. 2016; 53: 568-577https://doi.org/10.1597/15-036
      2. Shanbhag G., Pandey S., Mehta N., Kini Y., Kini A. A virtual noninvasive way of constructing a nasoalveolar molding plate for cleft babies, using intraoral scanners, CAD, and prosthetic milling. Cleft Palate-Craniofacial J. Published online 2019. doi:10.1177/1055665619886476.

        • Choi Y.S.
        • Shin H.S.
        Preoperative planning and simulation in patients with cleft palate using intraoral three-dimensional scanning and printing.
        J Craniofac Surg. 2019; 30: 2245-2248https://doi.org/10.1097/SCS.0000000000005983
        • Radojicic J.
        Cleft Care: intraoral 3D scanning.
        Cleft Palate Craniofac J. 2018; 55: 1330https://doi.org/10.1177/1055665617742806
        • Reichert F.
        • Amrhein P.
        • Uhlemann F.
        Unnoticed aspiration of palate plate impression material in a neonate: diagnosis, therapy, outcome.
        Pediatr Pulmonol. 2017; 52: E58-E60https://doi.org/10.1002/ppul.23710
        • Schober P.
        • Vetter T.R.
        Survival analysis and interpretation of time-to-event data: the tortoise and the hare.
        Anesth Analg. 2018; 127: 792-798https://doi.org/10.1213/ANE.0000000000003653
        • Chate R.A.
        A report on the hazards encountered when taking neonatal cleft palate impressions (1983-1992).
        Br J Orthod. 1995; 22: 299-307https://doi.org/10.1179/bjo.22.4.299
        • Dalessandri D.
        • Tonni I.
        • Laffranchi L.
        • et al.
        Evaluation of a digital protocol for pre-surgical orthopedic treatment of cleft lip and palate in newborn patients: a pilot study.
        Dent J. 2019; 7https://doi.org/10.3390/dj7040111
        • Gong X.
        • Dang R.
        • Xu T.
        • Yu Q.
        • Zheng J.
        Full digital workflow of nasoalveolar molding treatment in infants with cleft lip and palate.
        J Craniofac Surg. 2020; 31: 367-371https://doi.org/10.1097/SCS.0000000000006258
        • Weise C.
        • Frank K.
        • Wiechers C.
        • et al.
        Intraoral scanning of neonates and infants with craniofacial disorders: feasibility, scanning duration, and clinical experience.
        Eur J Orthod. 2021; 17 (Published online September)https://doi.org/10.1093/EJO/CJAB068
        • von Elm E.
        • Altman D.G.
        • Egger M.
        • Pocock S.J.
        • Gøtzsche P.C.
        • Vandenbroucke J.P.
        The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies.
        Lancet. 2007; 370: 1453-1457https://doi.org/10.1016/S0140-6736(07)61602-X
      3. ISO. Clinical investigation of medical devices for human subjects - Good clinical practice. Bs En Iso 14155-2:2009. Published 2011. Accessed March 17, 2022. https://www.iso.org/standard/71690.html

        • Harris P.A.
        • Taylor R.
        • Thielke R.
        • Payne J.
        • Gonzalez N.
        • Conde J.G.
        Research electronic data capture (REDCap)-A metadata-driven methodology and workflow process for providing translational research informatics support.
        J Biomed Inform. 2009; 42: 377-381https://doi.org/10.1016/j.jbi.2008.08.010
        • Harris P.A.
        • Taylor R.
        • Minor B.L.
        • et al.
        The REDCap consortium: building an international community of software platform partners.
        J Biomed Inform. 2019; : 95https://doi.org/10.1016/j.jbi.2019.103208
        • Nalabothu P.
        • Benitez B.K.
        • Dalstra M.
        • Verna C.
        • Mueller A.A.
        Three-dimensional morphological changes of the true cleft under passive presurgical orthopaedics in unilateral cleft lip and palate: a retrospective cohort study.
        J Clin Med. 2020; 9: 962https://doi.org/10.3390/jcm9040962
        • Benitez B.K.
        • Brudnicki A.
        • Surowiec Z.
        • et al.
        Continuous circular closure in unilateral cleft lip and plate repair in one surgery.
        J Cranio-Maxillofacial Surg. 2022; 50: 76-85https://doi.org/10.1016/j.jcms.2021.07.002
        • Brudnicki A.
        • Regulski P.A.
        • Sawicka E.
        • Fudalej P.S.
        Alveolar volume following different timings of secondary bone grafting in patients with unilateral cleft lip and palate. A pilot study.
        J Clin Med. 2021; 10https://doi.org/10.3390/jcm10163524
      4. Poets C.F., Wiechers C., Koos B., Muzaffar A.R., Gozal D. Pierre Robin and breathing: what to do and when? Pediatr Pulmonol. Published online 2021. doi:10.1002/ppul.25317.

        • Shen C.
        • Yao C.A.
        • Magee W.
        • Chai G.
        • Zhang Y.
        Presurgical nasoalveolar molding for cleft lip and palate: the application of digitally designed molds.
        Plast Reconstr Surg. 2015; 135: 1007e-1015ehttps://doi.org/10.1097/PRS.0000000000001286
        • Ayoub A.F.
        • Garrahy A.
        • Hood C.
        • et al.
        Validation of a vision-based, three-dimensional facial imaging system.
        Cleft Palate-Craniofacial J. 2003; 40: 523-529https://doi.org/10.1597/02-067
        • Ayoub A.
        • Khan A.
        • Aldhanhani A.
        • et al.
        The validation of an innovative method for 3D capture and analysis of the nasolabial region in cleft cases.
        Cleft Palate-Craniofacial J. 2021; 58: 98-104https://doi.org/10.1177/1055665620946987
        • Sharma V.P.
        • Bella H.
        • Cadier M.M.
        • Pigott R.W.
        • Goodacre T.E.E.
        • Richard B.M.
        Outcomes in facial aesthetics in cleft lip and palate surgery: a systematic review.
        J Plast Reconstr Aesthetic Surg. 2012; 65: 1233-1245https://doi.org/10.1016/j.bjps.2012.04.001
        • Zarean Parichehr
        • Zarean Paridokht
        • Thieringer Florian M.
        • Mueller Andreas A.
        • Kressmann Sabine
        • Erismann Martin
        • Sharma Neha
        • Benitez Benito K.
        A Point-of-Care Digital Workflow for 3D Printed Passive Presurgical Orthopedic Plates in Cleft Care.
        Children. 2022;