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1.
Cleft Palate Craniofac J ; : 10556656241239459, 2024 Mar 15.
Artigo em Inglês | MEDLINE | ID: mdl-38490221

RESUMO

OBJECTIVE: To investigate whether flexible nasopharyngoscopy, when performed in addition to magnetic resonance imaging (MRI), influences the type of surgery selected or success of surgery in patients with velopharyngeal insufficiency (VPI). DESIGN: Cohort study. SETTING: A metropolitan children's hospital. PATIENTS: Patients with non-syndromic, repaired cleft palate presenting for management of VPI. INTERVENTIONS: MRI and nasopharyngoscopy or MRI alone for preoperative imaging of the velopharyngeal mechanism. MAIN OUTCOME MEASURES: (1) Surgical selection and (2) resolution of hypernasality. All speech, MRI, and nasopharyngoscopy measurements were performed by raters blinded to patients' medical and surgical history. RESULTS: Of the 25 patients referred for nasopharyngoscopy, 76% completed the exam. Of the 41 patients referred for MRI, the scan was successfully completed by 98% of patients. Completion of nasopharyngoscopy was significantly (p=0.01) lower than MRI. Surgical selection did not significantly differ (p=0.73) between the group receiving MRI and nasopharyngoscopy and the group receiving MRI alone, nor was there a significant difference between these groups in the proportion of patients achieving resolution of hypernasality postoperatively (p=0.63). Percent total velopharyngeal closure assessments on nasopharyngoscopy and MRI were strongly correlated (r=0.73). CONCLUSIONS: In patients receiving MRI as part of their preoperative VPI evaluation, the addition of nasopharyngoscopy did not result in a difference in surgical selection or resolution of hypernasality. Routine inclusion of nasopharyngoscopy may not be necessary for the evaluation of velopharyngeal anatomy when MRI is available.

2.
mBio ; 15(2): e0306523, 2024 Feb 14.
Artigo em Inglês | MEDLINE | ID: mdl-38190129

RESUMO

Influenza A virus infection during pregnancy can cause adverse maternal and fetal outcomes but the mechanism responsible remains elusive. Infection of outbred mice with 2009 H1N1 at embryonic day (E) 10 resulted in significant maternal morbidity, placental tissue damage and inflammation, fetal growth restriction, and developmental delays that lasted through weaning. Restriction of pulmonary virus replication was not inhibited during pregnancy, but infected dams had suppressed circulating and placental progesterone (P4) concentrations that were caused by H1N1-induced upregulation of pulmonary cyclooxygenase (COX)-1-, but not COX-2-, dependent synthesis and secretion of prostaglandin (PG) F2α. Treatment with 17-α-hydroxyprogesterone caproate (17-OHPC), a synthetic progestin that is safe to use in pregnancy, ameliorated the adverse maternal and fetal outcomes from H1N1 infection and prevented placental cell death and inflammation. These findings highlight the therapeutic potential of progestin treatments for influenza during pregnancy.IMPORTANCEPregnant individuals are at risk of severe outcomes from both seasonal and pandemic influenza A viruses. Influenza infection during pregnancy is associated with adverse fetal outcomes at birth and adverse consequences for offspring into adulthood. When outbred dams, with semi-allogenic fetuses, were infected with 2009 H1N1, in addition to pulmonary virus replication, lung damage, and inflammation, the placenta showed evidence of transient cell death and inflammation that was mediated by increased activity along the arachidonic acid pathway leading to suppression of circulating progesterone. Placental damage and suppressed progesterone were associated with detrimental effects on perinatal growth and developmental delays in offspring. Treatment of H1N1-infected pregnant mice with 17-OHPC, a synthetic progestin treatment that is safe to use in pregnancy, prevented placental damage and inflammation and adverse fetal outcomes. This novel therapeutic option for the treatment of influenza during pregnancy should be explored clinically.


Assuntos
Vírus da Influenza A Subtipo H1N1 , Vírus da Influenza A , Influenza Humana , Gravidez , Feminino , Camundongos , Animais , Humanos , Progesterona/farmacologia , Placenta , Inflamação , Feto/metabolismo
3.
J Speech Lang Hear Res ; 67(1): 49-58, 2024 Jan 08.
Artigo em Inglês | MEDLINE | ID: mdl-37983129

RESUMO

OBJECTIVE: The purpose of this study was to identify infant and maternal factors associated with reduced human milk feeding among infants in a neonatal intensive care unit (NICU) with cleft lip with or without cleft palate (CL ± P). METHOD: Data collected on mothers and infants admitted to the NICU with CL ± P from the 2018 National Vital Statistics System were used for this study. Chi-square tests of independence and independent-samples t tests were used to compare categorical variables and continuous variables, respectively, among two groups of infants admitted to the NICU with CL ± P-those who did and did not receive human milk feeding at discharge. RESULTS: The sample included 660 infants admitted to the NICU with CL ± P, of which 353 received human milk at discharge. Significant differences were found between the two groups for marital status, mother's education, maternal smoking record, total number of prenatal visits, multiparity record, gestational age, birth weight, and use of assisted ventilation. CONCLUSIONS: Results indicated that, as a function of human milk feeding at discharge, mothers and their infants admitted to the NICU with CL ± P exhibited differences across infant and maternal factors. These findings further our understanding of this sample of mothers and infants with CL ± P while potentially identifying determinants to human milk feeding. This study provides insight into infant and maternal characteristics that may be associated with barriers to human milk feeding.


Assuntos
Fenda Labial , Fissura Palatina , Recém-Nascido , Lactente , Feminino , Gravidez , Humanos , Leite Humano , Aleitamento Materno/métodos , Unidades de Terapia Intensiva Neonatal , Recém-Nascido de muito Baixo Peso , Mães
4.
Magn Reson Med ; 91(1): 61-74, 2024 01.
Artigo em Inglês | MEDLINE | ID: mdl-37677043

RESUMO

PURPOSE: To improve the spatiotemporal qualities of images and dynamics of speech MRI through an improved data sampling and image reconstruction approach. METHODS: For data acquisition, we used a Poisson-disc random under sampling scheme that reduced the undersampling coherence. For image reconstruction, we proposed a novel locally higher-rank partial separability model. This reconstruction model represented the oral and static regions using separate low-rank subspaces, therefore, preserving their distinct temporal signal characteristics. Regional optimized temporal basis was determined from the regional-optimized virtual coil approach. Overall, we achieved a better spatiotemporal image reconstruction quality with the potential of reducing total acquisition time by 50%. RESULTS: The proposed method was demonstrated through several 2-mm isotropic, 64 mm total thickness, dynamic acquisitions with 40 frames per second and compared to the previous approach using a global subspace model along with other k-space sampling patterns. Individual timeframe images and temporal profiles of speech samples were shown to illustrate the ability of the Poisson-disc under sampling pattern in reducing total acquisition time. Temporal information of sagittal and coronal directions was also shown to illustrate the effectiveness of the locally higher-rank operator and regional optimized temporal basis. To compare the reconstruction qualities of different regions, voxel-wise temporal SNR analysis were performed. CONCLUSION: Poisson-disc sampling combined with a locally higher-rank model and a regional-optimized temporal basis can drastically improve the spatiotemporal image quality and provide a 50% reduction in overall acquisition time.


Assuntos
Imageamento por Ressonância Magnética , Fala , Imageamento por Ressonância Magnética/métodos , Processamento de Imagem Assistida por Computador/métodos , Algoritmos
5.
Cleft Palate Craniofac J ; : 10556656231215141, 2023 Nov 30.
Artigo em Inglês | MEDLINE | ID: mdl-38037274

RESUMO

OBJECTIVE: The purpose of this study was to determine the anatomical differences among selected individuals with a cleft palate repaired using "The Buccal Flap Approach" during primary palatoplasty compared to aged-matched participants without cleft palate. DESIGN: Observational, prospective. SETTING: Two regional hospitals. PARTICIPANTS: A total of 30 adult males consisting of 15 adults born with cleft palate who received the Double Opposing Z-Plasty plus Buccal Flaps (DOZP + BF) repair at the time of primary palatoplasty and no history of secondary speech surgery or orthognathic surgery and 15 adults without a history of cleft palate. INTERVENTIONS: All participants underwent MRI to visualize anatomy. MAIN OUTCOME MEASURES: Ten velopharyngeal and craniofacial anatomical measures. RESULTS: No statistically significant differences between groups were observed for velar thickness, velar length, pharyngeal depth, NSBa angle, SNB angle, or levator veli palatini length. Individuals with the DOZP + BF presented with a greater effective velar length (p < .001), greater effective VP ratio (p < .001), smaller SNA angle (p < .001), and smaller maximal velar stretch (p < .001) compared to the control participants. CONCLUSIONS: This study suggests that adult males who received the DOZP + BF repair at the time of primary palatoplasty and no history of secondary speech surgery or orthognathic surgery present with a longer effective velar length and larger effective VP ratio in comparison to the non-cleft group. Future research is needed to compare patients with and without favorable outcomes from multiple surgical types to fully understand how surgical techniques alter the anatomy.

6.
Plast Reconstr Surg Glob Open ; 11(11): e5375, 2023 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-37928635

RESUMO

Background: Secondary Furlow (Furlow) and buccal myomucosal flaps (BMMF) treat velopharyngeal insufficiency by lengthening the palate and retropositioning the levator veli palatini muscles. The criteria for choosing one operation over the other remain unclear. Methods: A single-center retrospective cohort study was conducted. Thirty-two patients with nonsyndromic, repaired cleft palate were included. All patients underwent a Furlow or BMMF. Outcome measures included (1) resolution of hypernasality 12 months postoperatively, (2) degree of improvement of hypernasality severity; and (3) change in velar length, as measured on magnetic resonance imaging scans obtained preoperatively and 12 months postoperatively. All measures were performed by raters blinded to participants' medical and surgical history. Results: Hypernasality was corrected to normal in 80% of the Furlow group and in 56% of the BMMF group. Patients receiving BMMF had more severe hypernasality during preoperative speech evaluation. Both groups had a median decrease of two scalar rating points for severity of hypernasality (P = 0.58). On postoperative magnetic resonance imaging, patients who underwent Furlow had a median increased velar length of 6.9 mm. Patients who received BMMF had a median increased velar length of 7.5 mm. There was no statistically significant difference between groups regarding increase in velar length (P = 0.95). Conclusions: Furlow and BMMF procedures increase velar length with favorable speech outcomes. The same degree of improvement for hypernasality was observed across groups, likely explained by the similar increase in velar length achieved. Anatomic changes in palate length and levator veli palatini retropositioning persist 1 year after surgery.

7.
J Speech Lang Hear Res ; 66(12): 4828-4837, 2023 Dec 11.
Artigo em Inglês | MEDLINE | ID: mdl-37902502

RESUMO

OBJECTIVE: Understanding the normal anatomy of velopharyngeal (VP) mechanism and the emergence of sexual dimorphism provides valuable insights into differences of VP anatomy among males and females. The purpose of this study is to examine sex differences in VP anatomy in a large data set of 3,248 9- and 10-year-old children. METHOD: Static three-dimensional magnetic resonance imaging was used to compare five VP characteristics including velar length, velar thickness, effective velar length, levator veli palatini muscle length, and pharyngeal depth between age-matched males (n = 1,670) and females (n = 1,578). Additionally, these dimensions were used to determine the VP ratio and effective VP ratio. RESULTS: Males showed significantly larger dimensions for all VP distances and significantly lower ratios of velar length and effective velar length to pharyngeal depth (p < .05). The magnitude of these effect sizes was small to medium, with Cohen's d values ranging from 0.12 to 0.63. Additionally, the VP ratio and effective VP ratio are lower among males compared to females (p < .05). CONCLUSIONS: Results suggest the presence of sexual dimorphism in the VP mechanism among 9- and 10-year-old children. These findings emphasize the necessity of using different normative data for males and females when making comparisons to patients with cleft palate.


Assuntos
Fissura Palatina , Insuficiência Velofaríngea , Criança , Humanos , Masculino , Feminino , Caracteres Sexuais , Palato Mole/fisiologia , Faringe/diagnóstico por imagem , Faringe/fisiologia , Imageamento por Ressonância Magnética/métodos
8.
bioRxiv ; 2023 Nov 12.
Artigo em Inglês | MEDLINE | ID: mdl-37745453

RESUMO

Influenza A virus infection during pregnancy can cause adverse maternal and fetal outcomes, but the mechanism responsible remains elusive. Infection of outbred mice with 2009 H1N1 at embryonic day (E) 10 resulted in significant maternal morbidity, placental tissue damage and inflammation, fetal growth restriction, and developmental delays that lasted through weaning. Restriction of pulmonary virus replication was not inhibited during pregnancy, but infected dams had suppressed circulating and placental progesterone (P4) concentrations that were caused by H1N1-induced upregulation of pulmonary cyclooxygenase (COX)-1, but not COX-2-, dependent synthesis and secretion of prostaglandin (PG) F2α. Treatment with 17-α-hydroxyprogesterone caproate (17-OHPC), a synthetic progestin that is safe to use in pregnancy, ameliorated the adverse maternal and fetal outcomes from H1N1 infection and prevented placental cell death and inflammation. These findings highlight the therapeutic potential of progestin treatments for influenza during pregnancy.

9.
Cleft Palate Craniofac J ; : 10556656231194511, 2023 Aug 09.
Artigo em Inglês | MEDLINE | ID: mdl-37554050

RESUMO

OBJECTIVE: To evaluate the influence of common pediatric orthodontic appliances on velopharyngeal (VP) MRI and to compare MR image sequences to determine if sequence parameters impact the visibility of key VP structures commonly assessed in clinical VP MRI. DESIGN: Participants undergoing orthodontic treatment completed a VP MRI study. Level of distortion caused by orthodontic devices on 8 anatomical sites of interest and using variable MRI sequences was evaluated. SETTING: Single institution. PARTICIPANTS: Nineteen participants undergoing orthodontic treatment. MAIN OUTCOME: Level of distortion caused by metal artifacts and MR sequence used. RESULTS: The results of this study demonstrate that appliances such as hyrax palatal expanders and braces with stainless steel brackets are acceptable for a VP MRI, while class II corrector springs are not recommended. The HASTE MRI sequence with 2D imaging techniques should be utilized if the child has orthodontic devices, while FSE and 3D imaging techniques are not recommended. The presence of wire spring coils and molar bands are likely to not to interfere with the MRI evaluation. CONCLUSIONS: Findings from this study suggest that the presence of orthodontic appliances does not hinder visualization of all velopharyngeal structures during an MRI. Therefore, careful consideration must be made prior to disqualifying or recommending patients for VP MRI.

10.
J Clin Invest ; 133(20)2023 10 16.
Artigo em Inglês | MEDLINE | ID: mdl-37581940

RESUMO

SARS-CoV-2 infection during pregnancy is associated with severe COVID-19 and adverse fetal outcomes, but the underlying mechanisms remain poorly understood. Moreover, clinical studies assessing therapeutics against SARS-CoV-2 in pregnancy are limited. To address these gaps, we developed a mouse model of SARS-CoV-2 infection during pregnancy. Outbred CD1 mice were infected at E6, E10, or E16 with a mouse-adapted SARS-CoV-2 (maSCV2) virus. Outcomes were gestational age-dependent, with greater morbidity, reduced antiviral immunity, greater viral titers, and impaired fetal growth and neurodevelopment occurring with infection at E16 (third trimester equivalent) than with infection at either E6 (first trimester equivalent) or E10 (second trimester equivalent). To assess the efficacy of ritonavir-boosted nirmatrelvir, which is recommended for individuals who are pregnant with COVID-19, we treated E16-infected dams with mouse-equivalent doses of nirmatrelvir and ritonavir. Treatment reduced pulmonary viral titers, decreased maternal morbidity, and prevented offspring growth restriction and neurodevelopmental impairments. Our results highlight that severe COVID-19 during pregnancy and fetal growth restriction is associated with heightened virus replication in maternal lungs. Ritonavir-boosted nirmatrelvir mitigated maternal morbidity along with fetal growth and neurodevelopment restriction after SARS-CoV-2 infection. These findings prompt the need for further consideration of pregnancy in preclinical and clinical studies of therapeutics against viral infections.


Assuntos
COVID-19 , SARS-CoV-2 , Feminino , Gravidez , Animais , Camundongos , Idade Gestacional , Ritonavir/farmacologia , Ritonavir/uso terapêutico , Antivirais/farmacologia , Antivirais/uso terapêutico
11.
J Womens Health (Larchmt) ; 32(8): 858-864, 2023 08.
Artigo em Inglês | MEDLINE | ID: mdl-37585515

RESUMO

Biological sex and gender-based constructs contribute significantly to the diversity of disease outcomes and treatment responses across the life course. To promote research considering sex and gender, the National Institutes of Health (NIH) Office of Research on Women's Health (ORWH) launched the Specialized Centers of Research Excellence (SCORE) on sex differences program. The Career Enhancement Core (CEC) of the Johns Hopkins SCORE on Sex and Age Differences in Immunity to Influenza (SADII) partnered with the Foundation for Gender-Specific Medicine, which matched NIH funding to support seed grants. Over 3 years we awarded 12 (10 were women faculty) seed grants to early-stage investigators. One year after the award, the seed grant awardees highlighted their progress, including publications, grant applications, and abstracts. All awardees noted challenges with their progress related to the COVID-19 pandemic and supply chain delays and shared suggestions for improving the programming of the CEC. They also highlighted the multiple ways the awards had helped them gain pilot data toward larger grants, build collaborative relationships, and present at the annual SCORE symposium. We describe a model and evidence supporting a private-academic collaboration to support the careers of early-stage investigators conducting research related to sex and gender.


Assuntos
Pesquisa Biomédica , COVID-19 , Estados Unidos , Humanos , Masculino , Feminino , Pandemias , COVID-19/epidemiologia , National Institutes of Health (U.S.) , Organização do Financiamento , Atenção à Saúde
12.
Cleft Palate Craniofac J ; : 10556656231183385, 2023 Jun 19.
Artigo em Inglês | MEDLINE | ID: mdl-37335134

RESUMO

OBJECTIVE: To introduce a highly innovative imaging method to study the complex velopharyngeal (VP) system and introduce the potential future clinical applications of a VP atlas in cleft care. DESIGN: Four healthy adults participated in a 20-min dynamic magnetic resonance imaging scan that included a high-resolution T2-weighted turbo-spin-echo 3D structural scan and five custom dynamic speech imaging scans. Subjects repeated a variety of phrases when in the scanner as real-time audio was captured. SETTING: Multisite institution and clinical setting. PARTICIPANTS: Four adult subjects with normal anatomy were recruited for this study. MAIN OUTCOME: Establishment of 4-D atlas constructed from dynamic VP MRI data. RESULTS: Three-dimensional dynamic magnetic resonance imaging was successfully used to obtain high quality dynamic speech scans in an adult population. Scans were able to be re-sliced in various imaging planes. Subject-specific MR data were then reconstructed and time-aligned to create a velopharyngeal atlas representing the averaged physiological movements across the four subjects. CONCLUSIONS: The current preliminary study examined the feasibility of developing a VP atlas for potential clinical applications in cleft care. Our results indicate excellent potential for the development and use of a VP atlas for assessing VP physiology during speech.

13.
Cleft Palate Craniofac J ; : 10556656231161991, 2023 Mar 08.
Artigo em Inglês | MEDLINE | ID: mdl-36890699

RESUMO

To determine the sensitivity and specificity of velar notching seen on nasopharyngoscopy for levator veli palatini (LVP) muscle discontinuity and anterior positioning.Nasopharyngoscopy and MRI of the velopharynx were performed on patients with VPI as part of their routine clinical care. Two speech-language pathologists independently evaluated nasopharyngoscopy studies for the presence or absence of velar notching. MRI was used to evaluate LVP muscle cohesiveness and position relative to the posterior hard palate. To determine the accuracy of velar notching for detecting LVP muscle discontinuity, sensitivity, specificity, and positive predictive value (PPV) were calculated.A craniofacial clinic at a large metropolitan hospital. PARTICIPANTS: Thirty-seven patients who presented with hypernasality and/or audible nasal emission on speech evaluation and completed nasopharyngoscopy and velopharyngeal MRI study as part of their preoperative clinical evaluation.Among patients with partial or total LVP dehiscence on MRI, presence of a notch accurately identified discontinuity in the LVP 43% (95% CI 22-66%) of the time. In contrast, the absence of a notch accurately indicated LVP continuity 81% (95% CI 54-96%) of the time. The PPV for the presence of notching to identify a discontinuous LVP was 78% (95% CI 49-91%). The distance from the posterior edge of the hard palate to the LVP, known as effective velar length, was similar in patients with and without notching (median 9.8 mm vs 10.5 mm, P = 1.00).The observation of a velar notch on nasopharyngoscopy is not an accurate predictor of LVP muscle dehiscence or anterior positioning.

14.
bioRxiv ; 2023 Jun 25.
Artigo em Inglês | MEDLINE | ID: mdl-36993658

RESUMO

SARS-CoV-2 infection during pregnancy is associated with severe COVID-19 and adverse fetal outcomes, but the underlying mechanisms remain poorly understood. Moreover, clinical studies assessing therapeutics against SARS-CoV-2 in pregnancy are limited. To address these gaps, we developed a mouse model of SARS-CoV-2 infection during pregnancy. Outbred CD1 mice were infected at embryonic day (E) 6, E10, or E16 with a mouse adapted SARS-CoV-2 (maSCV2) virus. Outcomes were gestational age-dependent, with greater morbidity, reduced anti-viral immunity, greater viral titers, and more adverse fetal outcomes occurring with infection at E16 (3rd trimester-equivalent) than with infection at either E6 (1st trimester-equivalent) or E10 (2nd trimester-equivalent). To assess the efficacy of ritonavir-boosted nirmatrelvir (recommended for pregnant individuals with COVID-19), we treated E16-infected dams with mouse equivalent doses of nirmatrelvir and ritonavir. Treatment reduced pulmonary viral titers, decreased maternal morbidity, and prevented adverse offspring outcomes. Our results highlight that severe COVID-19 during pregnancy and adverse fetal outcomes are associated with heightened virus replication in maternal lungs. Ritonavir-boosted nirmatrelvir mitigated adverse maternal and fetal outcomes of SARS-CoV-2 infection. These findings prompt the need for further consideration of pregnancy in preclinical and clinical studies of therapeutics against viral infections.

15.
Magn Reson Med ; 89(2): 652-664, 2023 02.
Artigo em Inglês | MEDLINE | ID: mdl-36289572

RESUMO

PURPOSE: To enable a more comprehensive view of articulations during speech through near-isotropic 3D dynamic MRI with high spatiotemporal resolution and large vocal-tract coverage. METHODS: Using partial separability model-based low-rank reconstruction coupled with a sparse acquisition of both spatial and temporal models, we are able to achieve near-isotropic resolution 3D imaging with a high frame rate. The total acquisition time of the speech acquisition is shortened by introducing a sparse temporal sampling that interleaves one temporal navigator with four randomized phase and slice-encoded imaging samples. Memory and computation time are improved through compressing coils based on the region of interest for low-rank constrained reconstruction with an edge-preserving spatial penalty. RESULTS: The proposed method has been evaluated through experiments on several speech samples, including a standard reading passage. A near-isotropic 1.875 × 1.875 × 2 mm3 spatial resolution, 64-mm through-plane coverage, and a 35.6-fps temporal resolution are achieved. Investigations and analysis on specific speech samples support novel insights into nonsymmetric tongue movement, velum raising, and coarticulation events with adequate visualization of rapid articulatory movements. CONCLUSION: Three-dimensional dynamic images of the vocal tract structures during speech with high spatiotemporal resolution and axial coverage is capable of enhancing linguistic research, enabling visualization of soft tissue motions that are not possible with other modalities.


Assuntos
Imageamento por Ressonância Magnética , Fala , Imageamento por Ressonância Magnética/métodos , Imageamento Tridimensional/métodos , Idioma , Linguística
16.
Cleft Palate Craniofac J ; 60(2): 249-252, 2023 02.
Artigo em Inglês | MEDLINE | ID: mdl-34787476

RESUMO

BACKGROUND: Non-sedated MRI is gaining traction in clinical settings for visualization of the velopharynx in children with velopharyngeal insufficiency. However, the behavioral adaptation and training aspects that are essential for successful pediatric MRI have received limited attention. SOLUTION: We outline a program of behavioral modifications combined with patient education and provider training that has led to high success rates for non-sedated velopharyngeal MRI in children.


Assuntos
Imageamento por Ressonância Magnética , Insuficiência Velofaríngea , Criança , Humanos , Imageamento por Ressonância Magnética/métodos
17.
Cleft Palate Craniofac J ; 60(11): 1499-1504, 2023 11.
Artigo em Inglês | MEDLINE | ID: mdl-35695193

RESUMO

This clinical case study describes the velopharyngeal anatomy and physiology in a patient who presented with SATB2-associated syndrome (SAS) and velopharyngeal insufficiency (VPI) in the absence of an overt cleft palate. The clinical presentation, treatment, outcome, and the contribution of anatomical findings from MRI to surgical treatment planning for this rare genetic disorder, SAS, are described. This case study contributes to our current understanding of the anatomy and physiology of the velopharyngeal mechanism in an individual born with SAS and non-cleft VPI. It also details the changes following bilateral buccal myomucosal flaps in this patient.


Assuntos
Fissura Palatina , Proteínas de Ligação à Região de Interação com a Matriz , Procedimentos de Cirurgia Plástica , Insuficiência Velofaríngea , Humanos , Insuficiência Velofaríngea/diagnóstico por imagem , Insuficiência Velofaríngea/cirurgia , Insuficiência Velofaríngea/complicações , Fissura Palatina/diagnóstico por imagem , Fissura Palatina/cirurgia , Fissura Palatina/complicações , Retalhos Cirúrgicos , Imageamento por Ressonância Magnética , Resultado do Tratamento , Fatores de Transcrição
18.
Cleft Palate Craniofac J ; 60(3): 299-305, 2023 03.
Artigo em Inglês | MEDLINE | ID: mdl-34812064

RESUMO

OBJECTIVE: The objective of this study was to examine differences in human milk feeding outcomes among infants admitted to a neonatal intensive care unit (NICU) with and without cleft lip and palate (CLP). METHOD: Data were used from a sample of infants admitted to the NICU with and without CLP from the 2018 National Vital Statistics System. For baseline comparisons, Chi-square tests of independence were used to compare categorical variables, and independent samples t tests were used for continuous variables. Logistic regression models were performed to determine the odds of human milk feeding at discharge in infants admitted to the NICU with CLP. RESULTS: The total sample included 345,429 infants admitted to the NICU, of which 660 had CLP. Significant differences were found among the following variables when baseline comparisons were made between infants admitted to the NICU with and without CLP: mother's race, mother's education, maternal smoking record, childbirth delivery method, presence of maternal pre-pregnancy diabetes, five-minute APGAR score, multiparity record (having more than one baby at birth), gestational age, and gestational weight. After controlling for baseline differences, results indicated reduced odds of human milk feeding at discharge in infants admitted to the NICU with CLP compared to those without CLP (OR = .543; 95% CI.455,.648). CONCLUSION: Results suggest reduced odds of human milk feeding at discharge among infants admitted to the NICU with CLP compared to those without CLP. These findings emphasize the necessity of awareness and facilitation of human milk feeding in this population.


Assuntos
Fenda Labial , Fissura Palatina , Recém-Nascido , Feminino , Gravidez , Lactente , Humanos , Unidades de Terapia Intensiva Neonatal , Leite Humano
19.
Cleft Palate Craniofac J ; 60(7): 858-864, 2023 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-35306864

RESUMO

To evaluate rates of breast milk feeding among infants with cleft lip with or without cleft palate (CL ± P) enrolled in Medicaid compared to Private Insurance/Self-Pay.This was a population-based retrospective cohort study.The 2018 US National Vital Statistics System-Natality component (NVSS-N) was used to examine nationwide birth certificate data.Infants with cleft lip with or without cleft palate and either Medicaid or Private Insurance/Self-Pay were included.Breast milk feeding rates among infants with CL ± P, as a function of insurance status.Chi-square tests of independence revealed that of 896 infants with CL ± P and insured by Medicaid, 527 (58.8%) were breast milk fed at discharge. Of 865 infants with CL ± P and insured by Private Insurance/Self-Pay, 621 (71.8%) were breast milk fed at discharge. Using logistic regression models and controlling for baseline demographic differences, results indicated that infants with CL ± P in the Medicaid group had reduced odds of breast milk feeding compared to the Private Insurance/Self-Pay group (OR = 0.08; 95% CI 0.56, 0.96).Breast milk support services are often necessary for the initiation of breast milk feeding among infants with CL ± P. However, these resources are likely not as readily available for those enrolled in Medicaid. These results suggest that infants with CL ± P, enrolled in Medicaid, may experience reduced breast milk feeding rates due to limited resources to initiate breast milk feeding. Factors that may promote breast milk feeding among this population are discussed.


Assuntos
Fenda Labial , Fissura Palatina , Feminino , Lactente , Humanos , Leite Humano , Estudos Retrospectivos , Aleitamento Materno
20.
Cleft Palate Craniofac J ; 60(10): 1250-1259, 2023 10.
Artigo em Inglês | MEDLINE | ID: mdl-35575228

RESUMO

OBJECTIVE: Velopharyngeal dysfunction (VPD) associated with 22q11.2 deletion syndrome (22q11.2DS) has a complex etiology. This study had 3 aims: (1) assess differences in velopharyngeal and levator muscle configuration during rest versus sustained speech production (2) compare differences in velopharyngeal changes between children with and without 22q11.2DS (3) examine the relationship between adenoid thickness, pharyngeal depth, and velopharyngeal changes. DESIGN: Cross-sectional. METHODS: A total of 22 participants, 11 with 22q11.2DS and 11 controls with normal speech and velopharyngeal anatomy (ages 4-12 years), underwent nonsedated MRI at rest and during sustained /i/. Differences in velar and levator muscle contraction across the 2 different conditions were analyzed, using matched paired t-tests. Mean differences across participant groups were examined. Correlation analyses were also conducted. RESULTS: When comparing differences between rest and sustained phoneme production (aim 1), significant (P < .05) differences were noted for all velar and levator muscle variables. For differences in velopharyngeal changes between children with and without 22q11.2DS (aim 2), VP ratio and effective VP ratio were noted to be significantly different. Pharyngeal depth and adenoid thickness were correlated with velar and levator muscle change measures and ratios (aim 3). CONCLUSION: Results from this study provide quantitative in vivo measurements of the contracted levator muscle and velum in young children with 22q11.2DS. Results demonstrated that VP ratio and EVP ratio are significantly different between children with and without 22q11.2DS and that pharyngeal depth is a strong clinical determinant of VPD in children with 22q11.2DS.


Assuntos
Síndrome de DiGeorge , Humanos , Criança , Pré-Escolar , Fala/fisiologia , Estudos Transversais , Faringe/diagnóstico por imagem , Faringe/fisiologia , Imageamento por Ressonância Magnética/métodos
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