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Am J Emerg Med ; 49: 302-303, 2021 Nov.
Article En | MEDLINE | ID: mdl-34182274

Angiotensin converting enzyme inhibitor-induced angioedema is typically easily recognizable in the emergency department. Angioedema lateralizing to one side, however, is infrequently reported, rare, and has the same potential of progression to airway compromise. We present of a case of an 80-year-old man with angioedema of the lower lip that had regressed prior to significant progression of right sided angioedema of the tongue and oropharynx.

Angioedema/etiology , Angiotensin-Converting Enzyme Inhibitors/adverse effects , Aged, 80 and over , Angiotensin-Converting Enzyme Inhibitors/therapeutic use , Humans , Male , Tongue/abnormalities , Tongue/drug effects
BMJ Case Rep ; 13(5)2020 May 19.
Article En | MEDLINE | ID: mdl-32434877

A newborn girl was referred to the otolaryngology service after prenatal imaging showed a right mandibular mass. Physical examination revealed a 1-2 cm mass along the right mandible with the appearance of a vestigial oral cavity. Tissue resembling the vermillion and primitive tongue appeared innervated and moved in conjunction with oral movements. MRI and CT of the mandible after birth confirmed a partially ossified soft tissue mass of the right mandibular body, containing unerupted teeth. She was taken to the operating room at 6 months of age for mass excision and reconstruction. Postoperatively, she healed well and was feeding without difficulty. Craniofacial duplication, including duplication of stomatodeal structures or diprosopus, is a rare condition with a variety of phenotypes. In the case of suspected craniofacial duplication, associated syndromes should be ruled out and appropriate imaging employed to determine the extent of involvement of adjacent structures, which will ultimately guide surgical planning.

Craniofacial Abnormalities/surgery , Mandible/surgery , Mouth Abnormalities/surgery , Tongue/surgery , Computed Tomography Angiography , Craniofacial Abnormalities/diagnosis , Female , Humans , Infant , Magnetic Resonance Imaging , Mandible/abnormalities , Mandible/diagnostic imaging , Mouth Abnormalities/diagnostic imaging , Reconstructive Surgical Procedures , Tongue/abnormalities , Tongue/diagnostic imaging
J Craniofac Surg ; 31(4): e391-e393, 2020 Jun.
Article En | MEDLINE | ID: mdl-32168129

Syngnathia is a rare facial anomaly associated with neonatal problems, including a compromised airway, and the inability to take in solids and/or liquids. Syngnathia is included within the spectrum of oromandibular limb hypogenesis syndrome, an extremely rare condition characterized by varying degrees of congenital malformation involving the tongue, mandible, and limbs. In this report, we describe the case of a 41-day-old Caucasian female infant who was unable to open her mouth beginning at birth. The authors performed osteotomies to separate fused bone, and placed a bite block at the osteotomy sites to prevent bone fusion recurrence. At 2 years of follow-up patient remained with 14 mm of mouth opening.

Jaw Abnormalities/diagnostic imaging , Mouth Abnormalities/diagnostic imaging , Abnormalities, Multiple , Female , Humans , Infant , Jaw Abnormalities/surgery , Mandible/abnormalities , Mouth Abnormalities/surgery , Osteotomy , Tongue/abnormalities
Anim Sci J ; 91(1): e13338, 2020.
Article En | MEDLINE | ID: mdl-32219936

Defective appearances, including white spotting (WS), tongue defect (TD), and nipple defect (ND), in Japanese Black cattle potentially lead to economic losses to farmers in Japan. We estimated genetic parameters of defective appearances using 553,433 records of Japanese Black heifer calves housed in the Kagoshima Prefecture. Variance and covariance were estimated using the Gibbs sampling algorithm. The estimated heritability ranged from 0.29 for TD to 0.76 for WS. Percent breeding value (%BV) estimates indicated high variation in WS and ND among sires, reflecting higher heritability. Furthermore, there was a positive linear relationship between the %BV estimate of a sire and the mean incidence rate of each defect in his female offsprings. TD was positively associated with other defects. Therefore, genetic factors strongly affect the incidence of defective appearances in Japanese Black cattle.

Cattle/abnormalities , Cattle/genetics , Animals , Breeding/economics , Female , Japan , Tongue/abnormalities
Taiwan J Obstet Gynecol ; 59(2): 323-326, 2020 Mar.
Article En | MEDLINE | ID: mdl-32127158

OBJECTIVE: To present molecular cytogenetic characterization of mosaic supernumerary ring chromosome 8 which has trisomy of a region of chromosome 8p12-q21.13 associated with congenital hypoplasia of the tongue and review of the literature. CASE REPORT: A 27 year-old woman presented with congenital hypoplasia of the tongue. The chromosome karyotype of peripheral blood lymphocytes was detected by conventional cytogenetic analysis. The genome copy number variations were detected by SNP array. Conventional cytogenetic analysis of the peripheral blood revealed a karyotype of 47,XX,+mar[60]/46,XX[40]. SNP array revealed that there was a duplication of 45.2 Mb at arr[hg19] 8p12q21.13(36,013,636-81,263,140) × 2-3. CONCLUSION: With this study a patient involving mosaic trisomy 8p12-q21.13 along with clinical properties, is described and compared to previously reported cases involving a small supernumerary marker chromosome (sSMC) derived from chromosome 8.

Cytogenetic Analysis/methods , Genetic Markers/genetics , Tongue/abnormalities , Trisomy/diagnosis , Adult , Chromosomes, Human, Pair 8/genetics , DNA Copy Number Variations , Female , Genetic Counseling , Humans , Karyotype , Karyotyping , Mosaicism , Pregnancy , Ring Chromosomes , Trisomy/genetics
AJR Am J Roentgenol ; 214(5): 1008-1018, 2020 05.
Article En | MEDLINE | ID: mdl-32069081

OBJECTIVE. The purpose of this article is to discuss imaging techniques and provide a pictorial review of pediatric tongue lesions. CONCLUSION. Pediatric tongue lesions represent an often-overlooked collection of abnormalities with a variety of clinical and radiologic features. The location of these lesions places the patient at risk for airway compromise. Systematic imaging evaluation of pediatric tongue lesions can help the radiologist arrive at a correct and timely diagnosis, thus improving and expediting patient care.

Tongue Diseases/diagnostic imaging , Tongue/abnormalities , Child , Diagnosis, Differential , Humans
Surg Radiol Anat ; 42(1): 9-13, 2020 Jan.
Article En | MEDLINE | ID: mdl-31531711

Intestinal duplication in the tongue is a rare entity. Occurrence in the anterior part of the tongue is exceptional. We report an intestinal duplication in the tongue causing eating difficulties and discuss the accuracy of embryologic and histopathology knowledge as radiology. A transoral complete resection of the lesion was performed, without postoperative complications. There was no recurrence with a follow-up of 15 years.

Digestive System Abnormalities/diagnostic imaging , Digestive System Abnormalities/surgery , Tongue Diseases/diagnostic imaging , Tongue Diseases/surgery , Tongue/diagnostic imaging , Tongue/surgery , Colon , Digestive System Abnormalities/complications , Digestive System Abnormalities/pathology , Esophageal Mucosa , Female , Heart Defects, Congenital/complications , Humans , Infant, Newborn , Intestinal Mucosa , Tongue/abnormalities , Tongue/pathology , Tongue Diseases/congenital , Tongue Diseases/pathology
J Med Case Rep ; 13(1): 56, 2019 Mar 10.
Article En | MEDLINE | ID: mdl-30851733

BACKGROUND: The lingual frenum is a fold of mucous membrane connecting the ventral tongue to the floor of the mouth. In general, lingual frenum serves multiple roles; its main function is to support the tongue and aid in limiting its movement in different directions. Any anatomical or functional deficiency of lingual frenum may have an impact on tongue functions based on its severity. Historically, the absence of lingual frenum was linked to multiple genetic and developmental conditions such as infantile hypertrophic pyloric stenosis, non-syndromic ankyloglossia diseases, and Ehlers-Danlos syndromes and was never reported in otherwise healthy individuals. CASE PRESENTATION: We report the absence of lingual frenum in an otherwise healthy 21-year-old Middle Eastern woman diagnosed during a routine dental examination. CONCLUSION: To the best of our knowledge, this is the first case to be reported in the literature with similar clinical presentation. Even without a significant impact on tongue movement or speech, it is important for health practitioners to be aware of such conditions and evaluation steps for diagnosis and management.

Dental Care , Incidental Findings , Lingual Frenum/abnormalities , Female , Humans , Tongue/abnormalities , Young Adult
Cir. plást. ibero-latinoam ; 45(1): 57-66, ene.-mar. 2019. ilus
Article Es | IBECS | ID: ibc-182679

Introducción y Objetivo: El colgajo FAMM (colgajo músculo-mucoso de arteria facial) descrito en 1992 por Pribaz y col, es un colgajo intraoral basado en la arteria facial, compuesto por mucosa oral, submucosa, músculo bucinador, arteria facial y por el plexo venoso correspondiente y puede ser de base inferior (flujo anterógrado) o superior (flujo retrógrado). Es un colgajo versátil que se puede usar en la reconstrucción de defectos de múltiples localizaciones (paladar, lengua o suelo de boca). Con este trabajo pretendemos demostrar su utilidad en la reconstrucción de diferentes defectos intraorales. Material y Método: Presentamos 3 casos en lo que empleamos el colgajo FAMM para reconstrucción intraoral: un paciente con anquiloglosia cicatricial secuela de carcinoma de suelo de boca, un paciente con fístula de paladar secuela de resección tumoral, y un paciente con exposición intraoral de arco mandibular por osteonecrosis secundaria a bifosfonatos. Resultados: Todos los colgajos sobrevivieron al 100% y permitieron una cobertura estable y duradera, con ausencia de complicaciones mayores. Conclusiones: El colgajo FAMM permite la reconstrucción de defectos intraorales y periorales con tejido bien vascularizado y de idénticas características a las de la zona a reconstruir, con baja morbilidad de la zona donante, lo que lo convierte en una excelente opción reconstructiva para defectos de esta región anatómica

Background and Objective: The facial artery musculo-mucosal (FAMM) flap, first described by Pribaz et al. in 1992, is an intraoral flap based on the facial artery. It is composed of mucosa, submucosa, buccinator muscle and the facial artery along with its venous plexus. The design of the flap can be inferiorly-based and rely on antegrade blood flow or superiorly-based with retrograde flow. The FAMM flap is a versatile flap that can be used for the reconstruction of defects of multiple locations (palate, lips, tongue, and floor of the mouth). The purpose of this study is to demonstrate the utility of the FAMM flap in the reconstruction of different intraoral defects. Methods: In this article the authors present 3 cases in which the FAMM flap was used for intraoral reconstruction: one patient with a history of ankyloglossia sequelae of a squamous cell carcinoma of the floor of the mouth; one patient with a palatal fistula sequelae of tumor excision; and one patient with a biphosphonate-related osteonecrosis of the mandible. Results: All flaps survived and provided a stable and reliable coverage of the defect. There were no major complications. Conclusions: The FAMM flap is a well vascularized flap that replaces like with like tissue. Because of its low morbidity, low rate of complications and reliable results, the FAMM flap is an excellent option for reconstruction of small to moderate intra-oral defects

Introdução e Objetivo: O retalho FAMM (facial artery musculo-mucosal flap), descrito em 1992 por Pribaz et al., é um retalho intra-oral baseado na artéria facial. É composto por mucosa oral, submucosa, músculo bucinador, artéria facial e pelo plexo venoso correspondente, podendo basear-se inferior (fluxo anterógrado) ou superiormente (fluxo retrógrado). É um retalho versátil que pode ser utilizado nareconstrução de defeitos em múltiplas localizações (palato, lábio, língua, pavimento da boca). Com este trabalho pretende-se demonstrar a utilidade do retalho FAMM na reconstrução de diferentes defeitos intra-orais. Material e Métodos: Os autores apresentam 3 casos em que se utilizou o retalho FAMM para reconstrução intra-oral: um doente com anquiloglossia cicatricial sequelar de carcinoma do pavimento da boca; uma doente com uma fístula do palato sequela de excisão tumoral; e um doente com exposição intra-oral do arco mandibular anterior por osteonecrose secundária a bifosfonatos. Resultados: Todos os retalhos sobreviveram a 100% e permitiram uma cobertura estável e duradoura, na ausência de complicações major. Conclusão: O retalho FAMM permite a reconstrução de defeitos intra e peri-orais com tecido bem vascularizado e de características idênticas à zona a reconstruir, com baixa morbilidade da zona dadora, o que o torna uma excelente opção reconstrutiva para defeitos desta região anatómica

Humans , Male , Female , Middle Aged , Aged , Surgical Flaps/surgery , Mouth Mucosa/abnormalities , Mouth Mucosa/surgery , Mouth Floor/abnormalities , Palate/abnormalities , Palate/surgery , Tongue/abnormalities , Tongue/surgery , Mouth Neoplasms/radiotherapy , Ankyloglossia/complications
Plast Reconstr Surg ; 143(5): 1456-1465, 2019 05.
Article En | MEDLINE | ID: mdl-30789482

BACKGROUND: Numerous studies have proven the efficacy of mandibular distraction osteogenesis or tongue-lip adhesion in Robin sequence infants with upper airway obstruction. However, none has compared health-related quality of life outcomes. METHODS: In the present retrospective study, Robin sequence infants younger than 1 year, who underwent mandibular distraction osteogenesis or tongue-lip adhesion, were included (2006 to 2016). The infants' caregivers were asked to complete a questionnaire based on the Glasgow Children's Benefit Inventory. RESULTS: The response rate was 71 percent (22 of the 31 questionnaires; mandibular distraction osteogenesis, 12 of 15; and tongue-lip adhesion, 10 of 16) and median age at surgery was 24 days (range, 5 to 131 days). Median total Glasgow Children's Benefit Inventory scores after mandibular distraction osteogenesis and after tongue-lip adhesion were 21.9 (interquartile range, 9.4) and 26.0 (interquartile range, 37.5), respectively (p = 0.716), indicating an overall benefit from both procedures. Positive changes were observed in all subgroups emotion, physical health, learning, and vitality. In syndromic Robin sequence, both procedures demonstrated a lower positive change in health-related quality of life compared with isolated Robin sequence (p = 0.303). CONCLUSIONS: Both surgical procedures demonstrated an overall benefit in health-related quality-of-life outcomes, with no significant differences. The authors' findings contribute to the debate regarding the use of mandibular distraction osteogenesis versus tongue-lip adhesion in the surgical treatment of Robin sequence; however, studies evaluating health-related quality of life in larger Robin sequence cohorts are necessary to identify which procedure is likely to be best in each individual Robin sequence infant. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Lip/surgery , Mandible/surgery , Osteogenesis, Distraction/methods , Pierre Robin Syndrome/surgery , Quality of Life , Tongue/surgery , Caregivers/statistics & numerical data , Female , Humans , Infant , Infant, Newborn , Male , Mandible/abnormalities , Retrospective Studies , Surveys and Questionnaires/statistics & numerical data , Tissue Adhesions/etiology , Tongue/abnormalities , Treatment Outcome
Int J Pediatr Otorhinolaryngol ; 117: 167-170, 2019 Feb.
Article En | MEDLINE | ID: mdl-30579074

We report the case of a 37-week old newborn presenting on day 1 of life with an apparent congenital fusion of the tongue to the hard palate, consistent with Ankyloglossum Superius syndrome. Physical exam along with endoscopy showed apparent fusion of the floor of the mouth to the anterior hard palate displacing the tongue into the nasal cavity and obstructing the oral airway. The child was nasotracheally intubated and brought to the operating room for lysis of the fusion under binocular microscopy. We review the literature on this rare condition and provide an algorithm for evaluating the neonatal airway in the setting of congenital oral abnormalities.

Airway Obstruction/congenital , Airway Obstruction/surgery , Palate, Hard/abnormalities , Tongue/abnormalities , Congenital Abnormalities/diagnostic imaging , Congenital Abnormalities/surgery , Endoscopy , Humans , Infant, Newborn , Male , Palate, Hard/diagnostic imaging , Syndrome , Tongue/diagnostic imaging
Sleep Breath ; 23(1): 311-317, 2019 Mar.
Article En | MEDLINE | ID: mdl-30506267

PURPOSE: Despite a broad range of diagnostic methods, identifying the site of obstruction in the upper respiratory tract in patients with obstructive sleep apnea is not always simple and straightforward. With regard to this problem, we present our observations about the specific shape of the epiglottis in patients with obstruction at the level of the tongue base and/or epiglottis. METHODS: One hundred and forty consecutive drug-induced sleep endoscopy (DISE) video recordings of patients with polygraphy-verified obstructive sleep apnea were analyzed by three independent observers. We compared the levels of obstruction using the VOTE classification and the shape of the epiglottis, both as seen during the DISE investigation and in the awake state. We have calculated the interrater reliability for VOTE classification results and epiglottis shape evaluation by three different observers. RESULTS: Out of 140 patients, there were 52 (37.1%) with a flat epiglottis. Within this group, there were only 3 (6%) cases in which obstructions at the tongue base and/or epiglottis level were not found. In the group with normally convex and omega-shaped epiglottis, obstruction at the tongue base level was observed in 28 patients (31.8%); obstruction at the epiglottis level was observed in 5 patients (5.7%); and obstruction at both the epiglottis and tongue base level was observed in 3 patients (3.4%). Interrater reliability for VOTE classification was poor for V (ICC = 0.414) and good for O (ICC = 0.824), T (ICC = 0.775), and E (ICC = 0.852). Additionally, interrater reliability was excellent for epiglottis shape (ICC = 0.912). CONCLUSION: In patients with obstructive sleep apnea, examinations in the awake state and drug-induced sleep endoscopy both showed that in most cases of obstruction at epiglottis and/or tongue base, the epiglottis was flat, i.e., lacking the typical anterior convexity in its upper part. We assume that the change of its shape is a result of degeneration of suspensory apparatus that maintains the shape of the epiglottis and holds it in its position. This could contribute to the better identification of patients with a narrowing at this level, and in turn to better decisions regarding the choice of the most suitable treatment.

Epiglottis/abnormalities , Sleep Apnea, Obstructive/etiology , Adult , Aged , Airway Obstruction/diagnosis , Airway Obstruction/etiology , Airway Obstruction/surgery , Endoscopy , Epiglottis/surgery , Female , Humans , Male , Middle Aged , Polysomnography , Risk Factors , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/surgery , Tongue/abnormalities , Tongue/surgery
Int J Paediatr Dent ; 28(4): 380-389, 2018 Jul.
Article En | MEDLINE | ID: mdl-29767443

BACKGROUND: Although there is a lack of strong evidence for the association between ankyloglossia in newborns and impaired breastfeeding, screening for ankyloglossia using the Neonatal Tongue Screening Test (NTST) is mandated by law in Brazilian maternities. AIM: To assess the reliability and validity of the NTST. DESIGN: cohort study; baseline sample comprised 268 mother-newborn dyads. At follow-up, 169 mothers were contacted by telephone. Interviews with the mothers for data collection were performed up to 48 h and at 1-3 months after childbirth. Trained and calibrated personnel performed the oral examinations of the newborns. Thirty newborns were examined for inter-reproducibility assessment. RESULTS: Of the 268 newborns included, 212 had a lingual frenulum that could be visually inspected and their NTST scores ranged from zero to nine (mean = 2.0, ±2.0). Interexaminer reproducibility was acceptable (Intraclass correlation coefficient = 0.77). Internal consistency of the NTST was poor (Cronbach's alpha = 0.28). Construct validity was investigated through the association between NTST scores and difficulties in breastfeeding at baseline and follow-up, and infants' weight gain at follow-up (mean age 32 ± 6.7 days). No statistically significant associations were found. CONCLUSION: NTST is neither reliable nor valid for detecting ankyloglossia that may interfere with breastfeeding in newborns.

Ankyloglossia/diagnosis , Neonatal Screening/methods , Ankyloglossia/congenital , Ankyloglossia/pathology , Female , Humans , Infant, Newborn , Lingual Frenum/abnormalities , Lingual Frenum/pathology , Male , Reproducibility of Results , Tongue/abnormalities , Tongue/pathology
Glob Chang Biol ; 24(8): 3620-3628, 2018 08.
Article En | MEDLINE | ID: mdl-29808947

Warmer climates are predicted to increase bark beetle outbreak frequency, severity, and range. Even in favorable climates, however, outbreaks can decelerate due to resource limitation, which necessitates the inclusion of competition for limited resources in analyses of climatic effects on populations. We evaluated several hypotheses of how climate impacts mountain pine beetle reproduction using an extensive 9-year dataset, in which nearly 10,000 trees were sampled across a region of approximately 90,000 km2 , that was recently invaded by the mountain pine beetle in Alberta, Canada. Our analysis supports the hypothesis of a positive effect of warmer winter temperatures on mountain pine beetle overwinter survival and provides evidence that the increasing trend in minimum winter temperatures over time in North America is an important driver of increased mountain pine beetle reproduction across the region. Although we demonstrate a consistent effect of warmer minimum winter temperatures on mountain pine beetle reproductive rates that is evident at the landscape and regional scales, this effect is overwhelmed by the effect of competition for resources within trees at the site level. Our results suggest that detection of the effects of a warming climate on bark beetle populations at small spatial scales may be difficult without accounting for negative density dependence due to competition for resources.

Coleoptera/physiology , Alberta , Animals , Coleoptera/classification , Competitive Behavior , Environmental Monitoring , Hypertelorism , Intellectual Disability , Kyphosis , Megalencephaly , Models, Biological , Population Dynamics , Seasons , Temperature , Tongue/abnormalities , Trees