Thinking Out-of-the-Box: A Non-Standard Application of Standard Pulse-Oximetry and Standard Near-Infrared Spectroscopy in a COVID-19 Patient.

PURPOSE: Purpose of this report is to describe the feasibility of lingual pulse oximetry and lingual near-infrared spectroscopy (NIRS) in a COVID-19 patient to assess lingual tissue viability after several days of mechanical ventilation in the prone position. MATERIALS & METHODS: In a COVID-19 ICU-patient, the tongue became grotesquely swollen, hardened and protruding from the oral cavity after 20 h of mechanical ventilation uninterrupted in the prone position. To assess the doubtful viability of the tongue, pulse-oximetric hemoglobin O2-saturation (SpO2; Nellcor, OxiMax MAX-NI, Covidien, MA, USA) and NIRS-based, regional tissue O2-saturation measurements (rSO2; SenSmart, Nonin, MN, USA) were performed at the tongue. RESULTS: At the tongue, regular pulse-oximetric waveforms with a pulse-oximetric hemoglobin O2-saturation (SpO2) of 88% were recorded, i.e. only slightly lower than the SpO2 reading at the extremities at that time (90%). Lingual NIRS-based rSO2 measurements yielded stable tissue rSO2-values of 76-78%, i.e. values expected also in other adequately perfused and oxygenated (muscle-) tissues. CONCLUSION: Despite the alarming, clinical finding of a grotesquely swollen, rubber-hard tongue and clinical concerns on the adequacy of the tongue perfusion and oxygenation, our measurements of both arterial pulsatility (SpO2) and NIRS-based tissue oxygenation (rSO2) suggested adequate perfusion and oxygenation of the tongue, rendering non-vitality of the tongue, e.g. by lingual venous thrombosis, unlikely. To our knowledge, this is the first clinical report of lingual rSO2 measurement.

Tongue symptoms, suspension force and duration during operative laryngoscopy.

PURPOSE: Suspension laryngoscopy is a commonly performed procedure in otolaryngology. During the procedure, the laryngoscope applies direct force to the tongue. Postoperative tongue symptoms include pain, swelling, numbness, taste disturbance, and rarely motor deficits. Duration and magnitude of force applied have previously been associated with post-operative throat and tongue pain, respectively. We sought to correlate postoperative tongue symptoms with magnitude of force applied and/or duration of suspension and investigate any risk factors for tongue morbidity. MATERIALS AND METHODS: A sample of patients undergoing suspension laryngoscopy between 2015 and 2018 were prospectively recruited. Those with preexisting tongue symptoms, disease or surgery were excluded. Patients completed preoperative and postoperative questionnaires evaluating tongue swelling, numbness, motion and taste disturbance. Symptoms were subjectively scored on a visual scale from 0 to 10. Patient demographics, past medical and social history were also recorded. Intraoperative pressures were measured using a spring force scale, positioned between the suspension arm and Mayo stand. Initial and end suspension forces and duration of suspension were recorded. RESULTS: 120 patients met inclusion criteria, of which 63 completed both preoperative and postoperative questionnaires. 6 patients (9.5%) experienced postoperative tongue symptoms. Suspension force and duration of suspension were not significantly predictive of postoperative tongue symptoms. While all symptomatic patients were current or former cigarette smokers, smoking status was not found to be a statistically significant factor. CONCLUSIONS: Neither suspension forces nor duration of suspension were predictive of postoperative tongue morbidity. Further research is needed to evaluate the role of smoking status on postoperative tongue symptoms.

Copaiba oil-resin (Copaifera reticulata Ducke) modulates the inflammation in a model of injury to rats' tongues.

BACKGROUND: The regeneration of integrity and tissue homeostasis after injury is a fundamental property and involves complex biological processes fully dynamic and interconnected. Although there are medications prescribed to accelerate the process of wound healing by reducing the exaggerated inflammatory response, comes the need to search for different compounds of Amazonian biodiversity that can contribute to the acceleration of the healing process. Among these products, the copaiba oil-resin is one of the most prominent feature in this scenario, as they have been reported its medicinal properties. METHODS: Aiming to evaluate the anti-inflammatory and healing effect of copaiba oil-resin (Copaifera reticulata Ducke) in transfixing injury of rats' tongues first proceeded up the copaiba oil-resin oral toxicity test in 5 male mice to stipulate the therapeutic dose which was established at 200 mg/kg/day. Then it was induced transfixing injury in a total of 15 Wistar rats. The animals were randomly divided into three groups based on the treatment: control group, dexamethasone group and copaiba oil-resin group. After 7 days of treatment, histological slides stained with hematoxylin and eosin was prepared. Immunohistochemistry for CD68 (macrophage marker) was performed and analyzed by the cell counter Image J. RESULTS: The acute toxicity test showed that the oil-resin copal has low toxicity. Furthermore, copaiba oil-resin therapy modulates the inflammatory response by decreasing the chronic inflammatory infiltrate, edema and specifically the number of macrophages. CONCLUSIONS: The results indicate the potential of the Amazon region and showed up relevant because therapy with this extract modulates the inflammatory process.

Itchy Lip-Swollen Tongue Swollen Lip It's not Always an Allergy to Dental Materials; Angioederna- Possibly Life Threatening! You Might See This in Your Practice.

When providing dental treatment, the dental clinician (DC) is expected to be knowledgeable in recognition and emergency management of an allergic response. Common manifestations of localized allergic responses can present as a rash or hives (urticaria), running of the nose, and swelling of the lips, eyes, face, respiratory and gastrointestinal mucosa. More critically, there is the potential for breathing difficulties from laryngeal swelling. Airway obstruction can be potentially life threatening.' Anaphylactic shock can be another serious reaction. Although most of these allergic responses can be minimal and managed with an antihistamine (diphenhydramine), more serious allergic reactions (marked breathing difficulty, anaphylaxis) may require the use of an intramuscular injection of epinephrine. This case report describes a patient who had what appeared to be a common allergic response to some aspect of dental materials or treatment but was subsequently diagnosed with an allergic condition known as angioedema (AE) that had the potential to be non-responsive to dental office emergency medications. AE may be due to the more common histamine generated process as described below. However, if the patient's AE is due to alternate pathophysiology, the DC may not be able to manage breathing difficulties due to airway swelling, which could be rapidly fatal.

Electromyography findings in radiation-induced unilateral tongue immobility.

BACKGROUND: We used electromyography to characterize hypoglossal nerve function among radiation-treated head and neck cancer survivors with later onset unilateral tongue immobility. METHODS: Patients with unilateral tongue immobility without evidence of recurrent cancer were seen at a tertiary academic institution between February and September 2021. All patients were at least 2 years post-treatment with radiation therapy for head and neck squamous cell carcinoma. Participants were under annual surveillance and displayed no evidence of operative injury to the hypoglossal nerve. RESULTS: The median symptom-free interval for the 10 patients included in this study was 13.2 years (range 2-25 years). Myokymia alone was present in 3 of 10 patients, fibrillation potentials alone were present in 3 of 10 patients, and 1 subject displayed both fibrillation and myokymia. Three out of 10 patients had normal hypoglossal nerve function. DISCUSSION: These findings highlight how disparate mechanisms may underlie similar clinical presentations of radiation-induced neuromuscular dysfunction.

Familial adult-onset Pompe disease associated with unusual clinical and histological features.

The adult-onset form of Pompe disease had a wide clinical spectrum, ranging from asymptomatic patients with increased CK to muscle cramps and pain syndrome or rigid-spine syndrome. In addition clinical severity and disease progression are greatly variable. We report on a family with 3 siblings characterized by an unusual adult-onset Pompe disease including dysphagia and weakness of tongue, axial and limb-girdle muscles, in association with atypical globular inclusions in muscle fibres. Our study confirms the great clinical and histological variability of adult-onset Pompe disease and further supports the need of careful evaluation of bulbar function in patients affected by this pathology.

Tongue-tie, breastfeeding difficulties and the role of Frenotomy.

UNLABELLED: A greater emphasis on exclusive breastfeeding in recent years has re-ignited the historical debate over the role of ankyloglossia (tongue-tie) in infants with breastfeeding difficulties. Several prospective cohort studies and randomized control trials have been published in this area in the last 10-15 years. We critically evaluated recent evidence and attempted to answer two important clinical queries in this area, that is, (i) whether ankyloglossia is associated with breastfeeding difficulties and (ii) whether frenotomy helps mother-baby dyad in such setting? CONCLUSION: Neonates with tongue-tie are at increased risk for breastfeeding difficulties. An early recognition of this association by primary care provider and prompt referral to a lactation consultant is important. In cases with clearly documented breastfeeding difficulties, frenotomy often results in rapid improvement in symptoms.

Oro-facial manifestations in patients with eating disorders.

Studies have reported that the oral health status is jeopardized in patients with eating disorders. The aim was to review the oro-facial manifestations in patients with eating disorders. The address the focused question was "What is the oro-dental health status in patients with eating disorders?" MEDLINE/PubMed and Google Scholar databases were searched from 1948 to March 2012 using the following terms in various combinations: "Anorexia nervosa", "bulimia nervosa", "eating disorders", "dental", "oral health status". Letters to the editor, unpublished data and articles published in languages other than English were excluded. Dry lips, burning tongue and parotid gland swelling are common manifestations in patients with eating disorders as compared to medically healthy controls. The association of dental caries and periodontal disease in patients with eating disorders remains debatable. Temporomandibular disorders have also been reported to be more prevalent in patients with eating disorders as compared to healthy controls. A critical oral-dental examination during routine dental check-ups may reveal valuable information regarding the presence or absence of eating disorders in routine dental patients. This may be important information, updating the medical history, supporting the role of the physician.

Expanding the phenotype of late-onset Pompe disease: tongue weakness: a new clinical observation.

INTRODUCTION: Following the clinical observation of lingual weakness in 2 patients with late-onset Pompe disease (LOPD), tongue strength was assessed in 19 consecutive patients to determine the frequency and severity of this neurological sign. METHODS: Lingual strength was assessed using manual muscle testing; if weakness was present, severity was established as mild, moderate, or severe. RESULTS: All the patients exhibited lingual weakness, even 2 asymptomatic patients with a positive family history. Weakness was mild in 12 (63%), moderate in 6 (32%), and severe in 1 (5%). Dysarthria and/or dysphagia were observed or reported in 7 of 19 (37%) patients. CONCLUSIONS: Lingual weakness may be present as an axial sign of LOPD, even relatively early in the disease course, and may contribute to the differential diagnosis of this now treatable condition. Dysphagia and/or dysarthria may also occur. This finding further expands the phenotype of LOPD.

The broadening application of chemodenervation in X-linked dystonia-parkinsonism (Part II): an open-label experience with botulinum toxin-A (Dysport®) injections for oromandibular, lingual, and truncal-axial dystonias.

While the majority of chemodenervation clinics worldwide typically use botulinum toxins for the treatment of common conditions such as blepharopsams, cervical dystonia, limb dystonia, and spasticity, the unusually high concentration of X-linked dystonia-parkinsonism (XDP) has allowed us to collect and describe our experience in the use of botulinum toxin type A (BoNT-A) on rarer dystonic patterns. BoNT-A (Dysport®) was injected in a total 109 dystonias of XDP. Our cohort included: 50 cases in the oromandibular area (jaw opening: 32 cases, jaw closing: 12 cases, and jaw deviation: 6 cases); 35 cases in the lingual area (tongue protrusion: 27 cases and tongue curling: 8 cases); and, 24 cases in the truncal-axial area (flexor: 12 cases, extensor: 7 cases, and lateral-extensor: 5 cases). Interestingly, pain, often a nonprominent symptom of dystonias, was frequently reported in 40/50 XDP cases with oromandibular dystonia and 18/24 XDP cases with truncal-axial dystonia. All BoNT-A procedures were performed under electromyography guidance. A "high potency, low dilution" BoNT-A protocol was applied for oromandibular, lingual, cranial, cervical, and distal limb dystonias; whereas for dystonias of the abdominal, paraspinal, and proximal limb muscles, a "low potency, high dilution" BoNT-A injection protocol was applied. Outcomes measures included: the global dystonia rating scale (DRS) and pain visual analog scale (VAS) reduction at week 4; duration of BoNT-A effects; and, side effect profile. The median DRS score after 4 weeks was 3 ("substantial improvement") for oromandibular and lingual dystonias and 2 ("moderate improvement") for truncal-axial dystonias. Pain reduction was significantly reduced (75%-80% in oromandibular; 30%-80% in truncal-axial dystonias). The median duration of BoNT-A effect was 16 weeks for oromandibular, 12 weeks for lingual, and 11 weeks for truncal-axial dystonias. Compared to a generally safe and well-tolerated BoNT-A injections for truncal-axial dystonias, the oromandibular-lingual dystonias had the following frequency of adverse events: oromandibular--19% in jaw opening and 17% in jaw closing dystonias; lingual--19% in tongue protrusion and 13% in tongue curling dystonias. The most frequent adverse events were mouth dryness and dysphagia. Thus, BoNT-A injection working protocols may be adopted in XDP dystonia that adheres to cost minimization (e.g., lower dose end per selected muscle), while achieving some benefit, and potentially reduce the adverse event profile.

[Isolated palsy of the hypoglossal nerve complicating infectious mononucleosis]. / Paralysie isolée du nerf hypoglosse compliquant une mononucléose infectieuse.

INTRODUCTION: Neurological complications of infectious mononucleosis are rare. Various disorders have been described: meningitis, encephalitis, peripheral neuropathy. Isolated cranial nerve palsy has rarely been reported. OBSERVATION: A 16-year-old man was admitted for isolated and unilateral hypoglossal nerve palsy, four weeks after infectious mononucleosis. Cerebral MRI, cerebrospinal fluid study and electromyography were normal. IgM anti-VCA were positive. Two months later, without treatment, the tongue had almost fully recovered. CONCLUSION: To the best of our knowledge, only seven cases of isolated palsy of the hypoglossal nerve complicating infectious mononucleosis have been previously reported.

Isolated lingual dystonia : responding to anticholinergic treatment.

We report a 42 year-old woman with 6 month history of isolated lingual dystonia induced during speaking and was treated with anticholinergic drug. She experienced a marked improvement in her abnormal tongue movements. Primary lingual dystonia is a rare presentation.

The Effect of Streptococcus salivarius K12 on Halitosis: a Double-Blind, Randomized, Placebo-Controlled Trial.

This study was to evaluate the effect of Streptococcus salivarius K12 on tongue coating-associated halitosis. Twenty-eight subjects having tongue coating-associated halitosis were randomly divided into either a test or control group. For each of the 30 days, the test subjects sucked S. salivarius K12 tablet while the control subjects sucked placebo tablets. All the subjects did not take physical (tongue scraping) and chemical (antiseptic mouth-rinse) oral cavity pretreatment prior to use of the tablets. At baseline, and on the 1st, 7th, and 14th day after completing the course of tablets, the subjects were assessed for their organoleptic test (OLT) scores, volatile sulfur compound (VSC) levels, and tongue coating scores (TCS). During the course, all subjects kept their routine oral care habits without scraping their tongue coating. Plaque index, probing depth, and bleeding index were recorded at baseline and at the completion of the trial. On the 1st day following the end of tablet use, the OLT scores and VSC levels had significantly decreased in the test group when compared with the baseline values (P = 0.001 and P = 0.012). The TCS in the test group were also significantly decreased (P = 0.05). At days 7 and 14, the OLT scores in the test group were still significantly lower than the baseline levels (P = 0.006 and P = 0.039 respectively). However, there were no statistical differences with OLT, VSC, and TCS between the test group and the placebo group by analysis of multi-level regression model. The use of S. salivarius K12 did not have significant effect on halitosis with tongue coating cause when the tongue coating was not physically or chemically pre-treated, which implies removing tongue coating is required before Streptococcus salivarius K12 use.

Congenital insensitivity to pain with anhidrosis syndrome: A series from Jordan.

OBJECTIVES: To present the clinical picture, the associated complications and the genetic findings of Jordanian patients diagnosed with Congenital insensitivity to pain with anhidrosis (CIPA). PATIENTS AND METHODS: This is a retrospective study including 7 patients diagnosed with CIPA presenting to Jordan University Hospital neurology clinic between 2001 and 2017. RESULTS: Among five families, seven patients were diagnose with CIPA and followed for a period ranging from one month to 6 years. The initial symptom observed in all patients was high fever in the first few days after birth, decreased sensation to pain and decreased sweating were later noted. Poor weight gain, microcephaly and global developmental delay were present in most cases. All patients had tongue ulcerations. Fingers/toes ulcerations were present in 6/7 (86.0 %), hip joint dislocation in 3/7 (43.0 %), chronic arthritis and joint swelling in 6/7 (86.0 %), corneal ulcers in 4/7 (57.1 %) and kidney amyloidosis in 1/7 (13.0 %) of all patients. Death occurred in 4/7 (57.1 %) patients. Consanguinity was present in all families. Mutation analysis revealed three variants in NTRK1 gene. The frameshift (c.1860_1861insT; p.Pro621fs) mutation was common in our series. One patient carried a novel missense mutation (c.2170 G > A; p.Gly724Ser). The third missense mutation (C2125 G > T; p.Val709Leu) was reported in a previous study in one patient. CONCLUSION: This cohort reveals a severe CIPA phenotype necessitating thorough multidisciplinary care and follow up.

Tongue weakness is associated with respiratory failure in patients with severe Guillain-Barré syndrome.

OBJECTIVE: Swallowing impairment may worsen respiratory weakness and conduct to respiratory complications such as aspiration pneumonia in Guillain-Barré syndrome (GBS). We prospectively evaluate how tongue weakness could be associated to bulbar dysfunction and respiratory weakness in severe GBS patients. MEASUREMENTS AND MAIN RESULTS: Tongue strength, dysphagia and respiratory parameters were measured in 16 GBS patients at intensive care unit (ICU) admission and discharge and in seven controls. Tongue strength was decreased in the GBS patients compared with the controls. At admission, patients with dysphagia and those requiring mechanical ventilation (MV) had greater tongue weakness. All the patients with initial tongue strength <150 g required MV during ICU stay. Tongue strength correlated significantly with respiratory parameters. CONCLUSION: This study confirms the strong association between bulbar and respiratory dysfunction in GBS admitted to ICU. Tongue weakness may be present in GBS, especially during the phase of increasing paralysis, and resolves during the recovery phase. Tongue strength and indices of global and respiratory strength vary in parallel throughout the course of GBS. Further studies are needed to assess if, when used in combination with other respiratory tests, tongue strength measurement could contribute to identify patients at high risk for respiratory complications.

Ankyloglossia: facts and myths in diagnosis and treatment.

BACKGROUND: The objective of this study was to systematically review the diagnostic criteria, indications, and need for treatment of ankyloglossia (tongue-tie), as well as the various treatment options for patients in different age groups. METHODS: The MEDLINE databases and the Cochrane Library were searched according to well-defined criteria, resulting in 64 included articles. The evidence regarding the classifications of tongue-tie, epidemiologic data, inheritance, breastfeeding problems, impaired tongue mobility, speech disorders, malocclusion, gingival recessions, therapy, and complications due to surgery was analyzed in detail. RESULTS: Different classifications for ankyloglossia have been proposed but not uniformly accepted. Breastfeeding problems in neonates could be associated with a tongue-tie, but not enough controlled trials have been performed to identify an ideal treatment option. In children and adults with ankyloglossia, limitations in tongue mobility are present, but the individual degree of discomfort, as well as the severity of an associated speech problem, are subjective and difficult to categorize. There is no evidence supporting the development of gingival recessions because of ankyloglossia. Frenotomy, frenectomy, and frenuloplasty are the main surgical treatment options to release/remove an ankyloglossia. Because of the limited evidence available, no specific surgical method can be favored. CONCLUSIONS: The lack of an accepted definition and classification of ankyloglossia makes comparisons between studies almost impossible. Because almost no controlled prospective trials for surgical interventions in patients with tongue-ties are present in the literature, no conclusive suggestions regarding the method of choice can be made. It also remains controversial which tongue-ties need to be surgically removed and which can be left to observation.