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1. Timing of primary surgery for cleft palate

Author

Gamble, Carrol, Persson, Christina, Willadsen, Elisabeth, Albery, Liz, Soegaard Andersen, Helene, Zattoni Antoneli, Melissa, Appelqvist, Malin, Aukner, Ragnhild, Bodling, Pia, Bowden, Melanie, Brunnegård, Karin, Cairns, Gillian, Calladine, Samantha, Campbell, Linsay, Clayton-Smith, Jill, Cooper, Rachael, Conroy, Elizabeth, El-Angbawi, Ahmed, Kildegaard Emborg, Berit, Enfält Wikman, Josefin, Fitzpatrick, Beth, Fukushiro, Ana Paula, Guedes de Azevedo Bento Gonçalves, Cristina, Havstam, Christina, Hvistendahl, Anne Katherine, Dahl Jorgensen, Line, Klinto, Kristina, Berntsen Kvinnsland, Marit, Larham, Catriona, Lemvik, Jorunn, Leturgie, Louise, Liljerehn, Eva, Lodge, Natalie, Lohmander, Anette, McMahon, Siobhan, Mehendale, Felicity, Coracine Miguel, Haline, Moe, Marianne, Bogh Nielsen, Joan, Nyberg, Jill, Pedersen, Nina-Helen, Phippen, Ginette, Alvarez Piazentin-Penna, Silvia Helena, Patrick, Kathryn, Pliskin, Lindsay, Rigby, Lucy, Semb, Gunvor, Southby, Lucy, Sporre, Maria, Björkman Taleman, Ann-Sofie, Tangstad, Jorid, Kiemle Trindade, Inge Elly, Underwood, Imogen, van Eeden, Stephanie, Raud Westberg, Liisi, Williamson, Paula Ruth, Paciello Yamashita, Renata, Munro, Kevin, Walsh, Tanya, Shaw, William, Gamble, Carrol, Persson, Christina, Willadsen, Elisabeth, Albery, Liz, Soegaard Andersen, Helene, Zattoni Antoneli, Melissa, Appelqvist, Malin, Aukner, Ragnhild, Bodling, Pia, Bowden, Melanie, Brunnegård, Karin, Cairns, Gillian, Calladine, Samantha, Campbell, Linsay, Clayton-Smith, Jill, Cooper, Rachael, Conroy, Elizabeth, El-Angbawi, Ahmed, Kildegaard Emborg, Berit, Enfält Wikman, Josefin, Fitzpatrick, Beth, Fukushiro, Ana Paula, Guedes de Azevedo Bento Gonçalves, Cristina, Havstam, Christina, Hvistendahl, Anne Katherine, Dahl Jorgensen, Line, Klinto, Kristina, Berntsen Kvinnsland, Marit, Larham, Catriona, Lemvik, Jorunn, Leturgie, Louise, Liljerehn, Eva, Lodge, Natalie, Lohmander, Anette, McMahon, Siobhan, Mehendale, Felicity, Coracine Miguel, Haline, Moe, Marianne, Bogh Nielsen, Joan, Nyberg, Jill, Pedersen, Nina-Helen, Phippen, Ginette, Alvarez Piazentin-Penna, Silvia Helena, Patrick, Kathryn, Pliskin, Lindsay, Rigby, Lucy, Semb, Gunvor, Southby, Lucy, Sporre, Maria, Björkman Taleman, Ann-Sofie, Tangstad, Jorid, Kiemle Trindade, Inge Elly, Underwood, Imogen, van Eeden, Stephanie, Raud Westberg, Liisi, Williamson, Paula Ruth, Paciello Yamashita, Renata, Munro, Kevin, Walsh, Tanya, and Shaw, William

Abstract

BACKGROUND: Among infants with isolated cleft palate, whether primary surgery at 6 months of age is more beneficial than surgery at 12 months of age with respect to speech outcomes, hearing outcomes, dentofacial development, and safety is unknown. METHODS: We randomly assigned infants with nonsyndromic isolated cleft palate, in a 1:1 ratio, to undergo standardized primary surgery at 6 months of age (6-month group) or at 12 months of age (12-month group) for closure of the cleft. Standardized assessments of quality-checked video and audio recordings at 1, 3, and 5 years of age were performed independently by speech and language therapists who were unaware of the trial-group assignments. The primary outcome was velopharyngeal insufficiency at 5 years of age, defined as a velopharyngeal composite summary score of at least 4 (scores range from 0 to 6, with higher scores indicating greater severity). Secondary outcomes included speech development, postoperative complications, hearing sensitivity, dentofacial development, and growth. RESULTS: We randomly assigned 558 infants at 23 centers across Europe and South America to undergo surgery at 6 months of age (281 infants) or at 12 months of age (277 infants). Speech recordings from 235 infants (83.6%) in the 6-month group and 226 (81.6%) in the 12-month group were analyzable. Insufficient velopharyngeal function at 5 years of age was observed in 21 of 235 infants (8.9%) in the 6-month group as compared with 34 of 226 (15.0%) in the 12-month group (risk ratio, 0.59; 95% confidence interval, 0.36 to 0.99; P = 0.04). Postoperative complications were infrequent and similar in the 6-month and 12-month groups. Four serious adverse events were reported (three in the 6-month group and one in the 12-month group) and had resolved at follow-up. CONCLUSIONS: Medically fit infants who underwent primary surgery for isolated cleft palate in adequately resourced settings at 6 months of age were less likely to have velopharyngeal insuffici

Published
2023
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2. Treatment of active nasal fricatives substituting /s/ in young children with normal palatal function using motor-based intervention

Author

Kjellmer, Liselotte, Raud Westberg, Liisi, Lohmander, Anette, Kjellmer, Liselotte, Raud Westberg, Liisi, and Lohmander, Anette

Abstract

Purpose: The aim was to evaluate the effect of a motor-based, hierarchically structured intervention directed at active nasal fricatives substituting /s/ in young children with normal palatal function. Method: An experimental single-subject design was replicated across three children, aged 4–6 years, with normal palatal function, who substituted oral /s/ with active nasal fricatives. Treatment was performed weekly by a speech-language pathologist and included home training conducted by parents. Audio documented probes were registered regularly and /s/-production evaluated as oral or nasal. Result: All children achieved 98–100% oral production of /s/ in six probed linguistic contexts at treatment end and exhibited good maintenance at follow-up. The four-year-olds showed gradual or inconsistent response and slower progress, the six-year-old direct response and faster progress. Conclusion: The study provides preliminary evidence suggesting positive intervention effects for treating active nasal fricatives in children with normal palatal function. However, possible confounding effects such as maturation or repeated testing could not be ruled out; thus, results need to be replicated with increased experimental control. Nevertheless, the study adds to the currently meagre empirical evidence-base for the population. Individual treatment response and progress patterns were found and data suggests that the intervention may be beneficial from age 4.

Published
2021
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4. Mean GOSLON Yardstick Scores After 3 Different Treatment Protocols—A Long-term Study of Patients With Unilateral Cleft Lip and Palate

Author

Peterson, Petra, primary, Mars, Michael, additional, Gowans, Alan, additional, Larson, Ola, additional, Neovius, Erik, additional, Henningsson, Gunilla, additional, Andlin-Sobocki, Anna, additional, Pegelow, Marie, additional, Lemberger, Mathias, additional, Raud-Westberg, Liisi M., additional, and Karsten, Agneta L-A, additional

Published
2018
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6. Speech outcome in young children born with unilateral cleft lip and palate treated with one- or two-stage palatal repair and the impact of early intervention.

Author

Raud Westberg, Liisi, Höglund Santamarta, Lena, Karlsson, Jenny, Nyberg, Jill, Neovius, Erik, and Lohmander, Anette

Subjects
*CLEFT lip, *CLEFT palate, *ARTICULATION disorders, *CHI-squared test, *CONSONANTS, *LONGITUDINAL method, *RESEARCH funding, *SPEECH evaluation, *SPEECH therapy, *TREATMENT effectiveness, *EARLY medical intervention, *DATA analysis software, *DESCRIPTIVE statistics, *EVALUATION, *SURGERY
Abstract

Objectives: The aim of this study was to describe speech at 1, 1;6 and 3 years of age in children born with unilateral cleft lip and palate (UCLP) and relate the findings to operation method and amount of early intervention received. Methods: A prospective trial of children born with UCLP operated with a one-stage (OS) palatal repair at 12 months or a two-stage repair (TS) with soft palate closure at 3–4 months and hard palate closure at 12 months was undertaken (Scandcleft). At 1 and 1;6 years the place and manner of articulation and number of different consonants produced in babbling were reported in 33 children. At three years of age percentage consonants correct adjusted for age (PCC-A) and cleft speech errors were assessed in 26 of the 33 children. Early intervention was not provided as part of the trial but according to the clinical routine and was extracted from patient records. Results: At age 3, the mean PCC-A was 68% and 46% of the children produced articulation errors with no significant difference between the two groups. At one year there was a significantly higher occurrence of oral stops and anterior place consonants in the TS group. There were significant correlations between the consonant production between one and three years of age, but not with amount of early intervention received. Conclusions: The TS method was beneficial for consonant production at age 1, but not shown at 1;6 or 3 years. Behaviourally based early intervention still needs to be evaluated. [ABSTRACT FROM AUTHOR]

Published
2019
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7. Mean GOSLON Yardstick Scores After 3 Different Treatment Protocols—A Long-term Study of Patients With Unilateral Cleft Lip and Palate.

Author

Peterson, Petra, Larson, Ola, Neovius, Erik, Raud-Westberg, Liisi M., Karsten, Agneta L-A, Mars, Michael, Gowans, Alan, Henningsson, Gunilla, Andlin-Sobocki, Anna, Pegelow, Marie, and Lemberger, Mathias

Subjects
SKELETAL muscle, CLEFT lip, CLEFT palate, DENTAL arch, DENTAL casting, ENDOSCOPIC surgery, MEDICAL protocols, ORAL surgery, RESEARCH evaluation, STATISTICS, PLASTIC surgery, TREATMENT effectiveness, INTER-observer reliability, MEDICAL records, RETROSPECTIVE studies, SURGICAL site, ORTHOGNATHIC surgery, SURGERY
Abstract

Objectives: (1) To evaluate dental arch relationships, with the Great Ormond Street, London and Oslo (GOSLON) Yardstick, of participants with Unilateral cleft lip and palate (UCLP) and treated with 1-stage palatal closure with 3 different surgical protocols (2) to compare the mean GOSLON ratings with other CLP centers. Design: Retrospective study of medical charts and dental models. Setting: Karolinska University Hospital, Stockholm, Sweden. Participants: Eighty-seven patients with UCLP operated with 1-stage palatal repair. Thirty-five were operated with Veau-Wardill-Kilner (VWK) technique 1975 to 1986, 31 with minimal incision technique (MIT) from 1987 to 1997, and 21 according to MIT with muscle reconstruction (MITmr) 1998 to 2004. Interventions: Dental casts at ages 5 (n = 87), 7 to 8 (n = 27), 10 (n = 81), 16 (n = 61), and 19 (n = 35) years were rated by 10 assessors with the GOSLON Yardstick. Information of other interventions was retrieved from patients' charts. Main outcome measures: Mean GOSLON ratings. Results: A total of 82% of the participants were rated as having excellent to satisfactory outcome. Weighted κ statistics for the 10 assessors was good for inter-rater agreement and good/very good for intra-rater agreement. Conclusions: The mean GOSLON score in the Stockholm overall material at age 10 was 2.67. The VWK technique resulted in a greater need of orthognathic surgery than the MIT (P < .01). The MITmr did not produce better dental arch relationships than MIT at age 5 (P < .05). The best dental arch relationships were found in the MIT group at 10 years, mean 2.58, which is not significantly different from other centers with excellent outcome except Gothenburg and Vienna. [ABSTRACT FROM AUTHOR]

Published
2019
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10. Mean GOSLON Yardstick Scores After 3 Different Treatment Protocols-A Long-term Study of Patients With Unilateral Cleft Lip and Palate.

Author

Peterson P, Mars M, Gowans A, Larson O, Neovius E, Henningsson G, Andlin-Sobocki A, Pegelow M, Lemberger M, Raud-Westberg LM, and Karsten AL

Subjects
Adolescent, Child, Child, Preschool, Clinical Protocols, Dental Arch, Humans, London, Models, Dental, Retrospective Studies, Sweden, Treatment Outcome, Young Adult, Cleft Lip, Cleft Palate
Abstract

Objectives: (1) To evaluate dental arch relationships, with the Great Ormond Street, London and Oslo (GOSLON) Yardstick, of participants with Unilateral cleft lip and palate (UCLP) and treated with 1-stage palatal closure with 3 different surgical protocols (2) to compare the mean GOSLON ratings with other CLP centers., Design: Retrospective study of medical charts and dental models., Setting: Karolinska University Hospital, Stockholm, Sweden., Participants: Eighty-seven patients with UCLP operated with 1-stage palatal repair. Thirty-five were operated with Veau-Wardill-Kilner (VWK) technique 1975 to 1986, 31 with minimal incision technique (MIT) from 1987 to 1997, and 21 according to MIT with muscle reconstruction (MITmr) 1998 to 2004., Interventions: Dental casts at ages 5 (n = 87), 7 to 8 (n = 27), 10 (n = 81), 16 (n = 61), and 19 (n = 35) years were rated by 10 assessors with the GOSLON Yardstick. Information of other interventions was retrieved from patients' charts., Main Outcome Measures: Mean GOSLON ratings., Results: A total of 82% of the participants were rated as having excellent to satisfactory outcome. Weighted κ statistics for the 10 assessors was good for inter-rater agreement and good/very good for intra-rater agreement., Conclusions: The mean GOSLON score in the Stockholm overall material at age 10 was 2.67. The VWK technique resulted in a greater need of orthognathic surgery than the MIT ( P < .01). The MITmr did not produce better dental arch relationships than MIT at age 5 ( P < .05). The best dental arch relationships were found in the MIT group at 10 years, mean 2.58, which is not significantly different from other centers with excellent outcome except Gothenburg and Vienna.

Published
2019
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