Maxillomandibular Fusion Along with Fusion of Dorsal Surface of the Tongue to the Hard Palate-A Case Report.

RATIONALE: This case report presents a rare combination of congenital anomalies in an otherwise healthy male infant born at 36 weeks. The infant was diagnosed with congenital maxillomandibular synechia, ectrodactyly, and ankyloglossia superior syndrome (ASS). PATIENT CONCERNS: Inability to open the mouth completely, feeding challenges, and a cleft palate. The infant was stabilized through successful positive pressure ventilation via a face mask at birth and enteral feeding was initiated via a feeding gastrostomy. EXAMINATION: Diagnostic tests revealed a midline palatal cleft, hypoplastic jaws, persistent metopic suture, and a bony fusion at the midline. TREATMENT: Sectioning of the bony spur along the midline and achieving a mouth opening of 2 cm post-manipulation. The patient is under follow-up, with future treatment plans including cleft palate correction at 12 months and potential frontomandibular and lower jaw advancement depending on growth trajectories. TAKEAWAY LESSONS: This case underscores the complexity of managing multiple congenital anomalies and the need for individualized treatment plans.

Superior laryngeal artery originating from the lingual artery.

In most cases, the superior laryngeal artery (SLA) branches from the superior thyroid artery, which, in turn, leaves the external carotid artery. Few dissection studies found previously that the SLA could originate from the lingual artery. We report here probably the first evidence of such a rare anatomical variation found unilaterally in a retrospectively evaluated by computed tomography angiography adult male case. The left SLA left a suprahyoid coil of the lingual artery and continued over the greater hyoid horn to enter the larynx through the thyrohyoid membrane. On both sides, thyroid foramina were found, but only the right one used for the entry of the right SLA. Therefore, the rare SLA origin from the lingual artery can be documented on computed tomography angiograms, which could help during preoperative evaluations and prevent unwanted surgical complications.

Fetal Micro and Macroglossia: Defining Normal Fetal Tongue Size.

OBJECTIVES: Abnormal fetal tongue size is a phenotypic feature of various syndromes including Beckwith-Wiedemann, Pierre-Robin, oromandibular limb hypoplasia, chromosomal aberrations, etc. Current data regarding normal fetal tongue size are limited. Hence, micro/macroglossia are subjectively determined. The aim of the study was to construct a contemporary fetal tongue nomogram and to assess its clinical contribution. METHODS: A prospective cross-sectional study was performed in well dated, low risk, singleton pregnancies. Fetal tongues were measured by 5 trained sonographers. Highest quality images were selected. Intra- and interobserver variability was assessed. Tongue length, width, area, and circumference 1st to 99th centiles were calculated for each gestational week. Based on the normal tongue size charts, we created a Tongue Centile Calculator. RESULTS: Over 18 months, 664 tongue measurements were performed. A cubic polynomial regression model best described the correlation between tongue size and gestational age. The correlation coefficient (r2 ) was 0.934, 0.932, 0.925, and 0.953 for tongue length, width, area, and circumference, respectively (P < .001). Intra- and interobserver variability had high interclass correlation coefficients (>0.9). Using the new charts, we were able to identify 2 cases of macroglossia, subsequently diagnosed with Beckwith-Wiedemann, and 4 cases of microglossia, 3 associated with Pierre-Robin sequence, and 1 associated with persistent buccopharyngeal membrane. CONCLUSIONS: We present novel fetal tongue size charts from 13 to 40 weeks of gestation. Clinical application of these nomograms may be beneficial in the prenatal diagnosis of syndromes or malformations associated with abnormal fetal tongue size.

A Rare Case of Cleft Palate Associated With Tongue Hamartoma: A Case Report and Systematic Review.

INTRODUCTION: Palate development involves a genetic regulation through a complex molecular mechanism that may be disrupted by environmental factors, resulting in impaired fusion and cleft palate formation. An encounter with a case of cleft palate due to dorsal tongue hamartoma prompted us to perform this systematic review. OBJECTIVE: To review the clinical profile and management approach for a case with cleft palate and tongue hamartoma. DESIGN: A systematic literature search was conducted using keywords related to cleft palate and tongue hamartoma in PubMed, Scopus, MEDLINE, and Scielo databases through December 2021, with no time or language restrictions. PATIENTS, PARTICIPANTS: Studies reporting patients with cleft palate and tongue hamartoma were included. MAIN OUTCOME MEASURE(S): Information related to clinical profile, diagnostic tests, histopathology, management, and outcomes were extracted.Fourteen relevant publications were identified with 16 cases reported so far. Among them, thirteen patients were females (81.25%), and 3 were males (18.75%). The age of presentation varied from birth to 19 years. Oral-facial-digital syndrome (type II) was the most commonly associated syndrome.Congenital tongue hamartoma with cleft palate is a rare presentation, which can present as an isolated entity or part of a syndrome. Genetic evaluation is warranted, particularly for multiple hamartomatous lesions. The preferred treatment is immediate excision of hamartoma while following a standard timeline for palatoplasty.

Controversy Over Tongue-Tie: Divisions in the Community of Healthcare Professionals.

While recent decades have seen a rapid rise in cases of infant tongue-tie and in surgery to correct it, a controversy is now raging over the condition. Opinion is especially divided over so-called posterior tongue-tie, a variant which is detected based on the "feel" of the sub-lingual space. Drawing on ethnographic research with clinicians in England, we clarify the professional and personal commitments involved in the controversy. Our analysis is informed by Douglas' theory of cultural representations (grid-group theory), in which ideas of what is natural and unnatural constitute central metaphors.

Subglossopalatal Membrane With Associated Cleft Palate, Cardiovascular, and Neurologic Anomalies.

ABSTRACT: Subglossopalatal membrane (or subglossopalatal synechia) is a rare clinical entity that can lead to respiratory distress and feeding difficulty due to oral obstruction. Here, the authors present a case of subglossopalatal membrane with associated cleft palate and cardiovascular and neurologic anomalies that was treated with surgical excision and lip-tongue adhesion. Etiology of these membranes is believed to be intrauterine fetal insult. Membranes should be treated with excision, whereas taking care to ensure patency of the airway. Presence of a subglossopalatal membrane should prompt thorough examination for additional congenital anomalies.

Malformação venolinfática em borda lateral de língua: relato de caso / Venolymphatic malformation in lateral edge of the tongue: case report

Resumo As malformações vasculares são anomalias que podem acometer veias, vasos linfáticos e artérias de forma isolada ou mista. Quando se apresentam de forma mista, com componentes venosos e linfáticos, são denominadas malformação venolinfática ou linfático-venosa, de acordo com sua constituição predominante. Embora seja um distúrbio benigno de bom prognóstico, é localmente invasivo, podendo levar a deformidade e havendo, ainda, a propensão de recorrência local. O presente artigo traz um caso de malformação venolinfática com localização incomum em borda lateral de língua, abordando-se a conduta clínica e o referencial teórico vigente.

Abstract Vascular malformations are vascular anomalies that can affect veins, lymphatic vessels, and/or arteries in isolated or mixed form. When they present in the mixed form with venous and lymphatic involvement, they are called venolymphatic or lymphatic-venous malformations, depending on their predominant component. Although these are benign disorders with good prognosis, they are locally invasive and may lead to deformity, while there is also a propensity for local recurrence. This article presents a case of venolymphatic malformation with unusual localization on the lateral border of the tongue, addressing the clinical conduct and the current theoretical framework.

Unilateral angioedema.

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.

Scleroderma en Coup de Sabre, Parry-Romberg Hemifacial Atrophy and Associated Manifestations of the Eye, the Oral Cavity and the Teeth: A Danish Follow-Up Study of 35 Patients Diagnosed between 1975 and 2015.

BACKGROUND: Scleroderma en coup de sabre (ECDS) and Parry-Romberg idiopathic hemifacial atrophy (IHA) may affect the eyes, oral cavity, teeth and possibly the brain. OBJECTIVE: Systematic follow-up study of ECDS/IHA-associated manifestations including ophthalmic and dental status. METHODS: Medical records of ECDS and IHA patients diagnosed in a 40-year period (1975-2015) were reviewed, and patients were re-examined. RESULTS: Thirty-five patients were included. Twenty-two patients (63%) had ECDS and 4 patients (11%) IHA. In 9 cases (26%), ECDS and IHA were found in the same patient. The ipsilateral eye was affected in 9 patients (26%). Ipsilateral abnormalities of the teeth and the tongue were found in 13 (46%) out of 28 examined. Eleven (31%) had extrafacial scleroderma on the trunk or the extremities. Neurological findings were not verified as ECDS/IHA related. CONCLUSION: ECDS and IHA are related and often overlap with concomitant affections of the connective tissues of the face on the ipsilateral side. Ocular and dental abnormalities are common and follow the distribution of the primary affection, for example, a paramedian line in the front and segmental affection of the maxilla and the mandible. The affections point to predisposing dysmorphogenetic events in embryonal life affecting the face, with abnormality of crest cells at the stage when they migrate from behind over the scalp or laterally to the face to mix up with mesenchymal tissues of the frontonasal, maxillary and mandibular processes. The study emphasizes that routine evaluation of ECDS and IHA should include ophthalmological and dental specialist examinations.

The Relationship of Ankyloglossia With Gender in Children and the Ideal Timing of Surgery in Ankyloglossia.

INTRODUCTION: Ankyloglossia is a congenital condition that restricts tongue mobility. The aim of this study is to evaluate the relationship between gender and pediatric ankyloglossia and evaluate the planning of ideal timing of surgery. METHODS: The files of pediatric patients in the Turkish population treated surgically for tongue-tie between June 2014 to June 2018 were scanned retrospectively. RESULTS: Three hundred and eighty-two pediatric patients were included in the study. Of these, 115 (30.1%) were female and 267 (69.9%) were male. The prevalence of ankyloglossia was significantly higher in males than in females (P < .001). The age of the patients at time of surgery ranged from 1 day to 114 months. The most common indication was sucking/feeding difficulties (82%) in patients younger than 2 years, and the most common symptom was speech problems (67%) in patients aged 2 years and older. CONCLUSION: In our study, the prevalence of ankyloglossia in Turkish society was significantly higher in males. Frenectomy surgery is a safe procedure that can be performed on the first day of life in newborns.

An accessory tongue in a child: A clinical case.

A 20-month-old male patient presented with a tumor on his tongue. The tumor was detected at birth with an initial size of 5 mm; thereafter, it gradually grew larger. The patient had no symptoms, but he frequently touched the tumor, as he grew older. The authors performed a tumor resection under general anesthesia, and the histopathological diagnosis confirmed that it was an accessory tongue, with the same components as that of a normal tongue. An accessory tongue regularly grows bigger as a patient grows older, resulting in dysphagia and articulation disorder. Younger patients should get accessory tongues surgically resected as soon as they are old enough to receive general anesthesia.

Analysis of Tongue Color-Associated Features among Patients with PCR-Confirmed COVID-19 Infection in Ukraine

ABSTRACT Objective: To evaluate and systematize tongue color-related manifestations among patients with PCR-confirmed COVID-19 infection. Material and Methods: This retrospective study included analysis of tongue images obtained from patients with PCR-confirmed COVID-19 infection. Evaluation of coronavirus disease severity (mild, moderate, severe, critical) was provided, considering clinical symptomatology and results of laboratorial and instrumental diagnostic methods. Each picture was analyzed considering the parameters of color of the tongue and color of the tongue plaque by two dental specialists. Cochran-Armitage test for trend was used to evaluate associations between the tongue color and tongue plaque color, and coronavirus disease severity. Results: The most prevalent tongue colors were pale pink, red and dark red (burgundy color). A total of 64.29% of patients with mild disease demonstrated pale pink color of the tongue. Patients with moderate coronavirus disease were characterized with the adverse trend: 62.35% of them presented with red-colored tongue, while in 37.64% of cases, the tongue was pale pink. Severe COVID-19 patients, almost in 90% of the cases, had either red or burgundy color of the tongue. Conclusion: SARS-COV-2 infection is not manifested by tongue-targeted or tongue-specific signs and features; however, coronavirus disease itself provokes changes within the tongue color and tongue plaque color similar to those registered during other internal pathologies.

Duplication of the oral cavity and mandible: a rare congenital craniofacial anomaly.

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.

Molecular cytogenetic characterization of mosaicism for a small supernumerary marker chromosome derived from chromosome 8 associated with congenital hypoplasia of the tongue and review of the literature.

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.

Syngnathia in Spectrum of Oromandibular Limb Hypogenesis Syndrome.

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.

Estimation of genetic parameters of defective appearances in Japanese Black heifer calves in Kagoshima.

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.