Differing Progression to Posterior Glottic Stenosis in Autoimmune and Idiopathic Subglottic Stenosis.

OBJECTIVES/HYPOTHESIS: We sought to characterize rates of progression to posterior glottic stenosis (PGS) from autoimmune or idiopathic subglottic stenosis. STUDY DESIGN: This was a retrospective review. METHODS: Patients from a tertiary-care laryngology practice over a 10-year period with autoimmune or idiopathic subglottic stenosis (SGS) were included. Patients with a history of prolonged intubation or other causes of iatrogenic stenosis were excluded. PGS was confirmed on videostrobolaryngoscopy recordings by a fellowship-trained laryngologist. PGS type (1-4) was also recorded. Demographic information was recorded, and if applicable, autoimmune disease type was specified. Time until PGS was recorded along with the number of interventions. Chi-squared analysis was used to compare PGS in autoimmune and idiopathic SGS. RESULTS: A total of 77 patients were identified with autoimmune (32 patients) or idiopathic (45 patients) subglottic stenosis. Autoimmune pathologies included systemic lupus erythematosus, granulomatosis with polyangiitis (GPA), rheumatoid arthritis, relapsing polychondritis, and sarcoidosis, with GPA the most common (14/32). Patients with autoimmune SGS had a higher rate of PGS (10 of 32) compared to idiopathic subglottic stenosis (1 of 45) for an odds ratio of 20 (95% CI: 2.4-166.4, P = .006). Patients with idiopathic SGS were more likely to be female (all 45 compared to 29/32 autoimmune, P = .07) and older (mean 53 (range 29-75) compared to 46 (20-82), P = .02). CONCLUSIONS: In this large patient cohort, autoimmune SGS patients were found to have a higher likelihood of developing PGS compared to their idiopathic counterparts, suggesting that counseling for this progression may be warranted. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:1816-1820, 2021.

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.

Immunohistopathology of the Newly Discovered Giant Papillae Tongue Disorder in Organ-Transplanted Children.

BACKGROUND: Giant papillae tongue disorder (GPTD) is a newly discovered, long-lasting clinical disorder that may develop in organ-transplanted pediatric recipients. The key feature of this disorder is the unique tongue lesion, which comprises swollen fungiform papillae. The aim of this study was to characterize the immunohistopathology of this novel inflammatory condition. METHODS: Six organ transplanted children with GPTD were included in the study. Routine histopathology and immunohistochemical stainings for CD3, CD4, CD8, CD25, FOXP3, CD20, CD138, CD68, CD1a, CD15, CD23, and mast cell tryptase were performed. RESULTS: Immunohistochemical analyses of the oral lesions revealed a subepithelial infiltrate that was primarily composed of CD3- and CD4-positive T cells, CD20-expressing B cells, macrophages, and CD138-positive plasma cells. The CD20-positive cells did not display the typical B cell morphology, having in general a more dendritic cell-like appearance. The CD138-expressing plasma cells were distinctly localized as a dense infiltrate beneath the accumulation of T cells and B cells. Increased numbers of CD1a-expressing Langerhans cells were detected both in the epithelium and connective tissue. Because no granulomas were observed and only single lesional eosinophils were detected, GPTD does not resemble a granulomatous or eosinophilic condition. CONCLUSIONS: We describe for the first time the immunopathological characteristics of a novel inflammatory disorder of the oral cavity, which may develop after solid organ transplantation in children.

Epstein-Barr virus-positive mucocutaneous ulcer arising in a post-hematopoietic cell transplant patient followed by polymorphic posttransplant lymphoproliferative disorder and cytomegalovirus colitis.

Epstein-Barr virus (EBV)-positive mucocutaneous ulcer (EBVMCU) is a newly described entity occurring in elderly or iatrogenically immunocompromised patients. We describe a case of EBVMCU arising in a post-hematopoietic cell transplant patient and followed by EBV-positive polymorphic posttransplant lymphoproliferative disorder (EBV+ polymorphic PTLD). The patient, a 52-year-old woman, received chemotherapy and autologous peripheral blood stem cell transplantation for relapsed diffuse large B-cell lymphoma (DLBCL). She achieved complete remission and was followed up in an outpatient clinic after discharge. One year later, EBVMCU appeared in the tongue and exhibited spontaneous regression. Six months after the regression of the EBVMCU, she had EBV+ polymorphic PTLD, analogous to EBV+ polymorphic DLBCL. The therapy for PTLD was not effective, and the patient finally died of disease progression. This was the first case of EBVMCU characterized by both an association with autologous peripheral blood stem cell transplantation and subsequent emergence of malignant lymphoma in a patient with relapsed DLBCL.

Primary mucocutaneous histoplasmosis in an immunocompetent patient.

We report a case of primary mucocutaneous histoplasmosis in an immunocompetent individual. The patient, a 61-year-old woman, presented with a non-healing ulcer on the lateral border of her tongue. Excisional biopsy of the lesion was consistent with histoplasmosis and no evidence of pulmonary or disseminated infection was found. Although mucocutaneous infection has been well-described as a manifestation of disseminated disease, especially in immunocompromised individuals, oral infections in immunocompetent patients are rare.

IgG4-related sclerosing disease clinically mimicking oral squamous cell carcinoma.

IgG4-related sclerosing disease is a distinct clinicopathologic entity known to involve the maxillofacial region, particularly the salivary, lacrimal, and pituitary glands. We report a case with lesions involving the tongue and palatine tonsil with associated skin lesions. A 45-year-old female patient presented with a history of soreness, dysphagia, and an asymptomatic rash involving the upper trunk. The initial clinical diagnosis of her oral lesions was squamous cell carcinoma. The diagnosis of an IgG4-related lesion was confirmed by histologic examination of the oral and skin lesions as well as confirmation of raised serum IgG4 levels. Tapering systemic corticosteroid therapy resulted in complete resolution of the lesions. This is the first report of IgG4-related sclerosing disease presenting as concurrent oral and skin lesions, with the oral lesion clinically resembling oral squamous cell carcinoma. Such lesions present a diagnostic challenge, but the outcome is very favorable.

Eosinophilic ulcer: the role of stress-induced psychoneuroimmunologic factors.

INTRODUCTION: Eosinophilic ulcer (EU) is a rare pathology and its etiology is still slightly known. It is a benign lesion characterized by fast-growing ulceration with elevated and indurated borders, most commonly affecting the tongue. CASE REPORT: The authors describe a case of EU on a lingual border that was initiated and had its clinical behavior altered by the psychological stress the patient was experiencing. DISCUSSION: This paper discusses the stress effects that alter the individual's immunologic response, thus attracting mast cells and eosinophils towards the mucosal epithelium, which are involved in eosinophilic ulcer. The authors make an association between eosinophilic ulcer and atopic dermatitis, two diseases that appear to have a similar, though not fully defined, etiology. The psychological stress factor was considered a predisponent factor for eosinophilic ulcer etiology and its interference in the etiology and evolution of this disease should be considered.

Common tongue conditions in primary care.

Although easily examined, abnormalities of the tongue can present a diagnostic and therapeutic dilemma for physicians. Recognition and diagnosis require a thorough history, including onset and duration, antecedent symptoms, and tobacco and alcohol use. Examination of tongue morphology and a careful assessment for lymphadenopathy are also important. Geographic tongue, fissured tongue, and hairy tongue are the most common tongue problems and do not require treatment. Median rhomboid glossitis is usually associated with a candidal infection and responds to topical antifungals. Atrophic glossitis is often linked to an underlying nutritional deficiency of iron, folic acid, vitamin B12, riboflavin, or niacin and resolves with correction of the underlying condition. Oral hairy leukoplakia, which can be a marker for underlying immunodeficiency, is caused by the Epstein-Barr virus and is treated with oral antivirals. Tongue growths usually require biopsy to differentiate benign lesions (e.g., granular cell tumors, fibromas, lymphoepithelial cysts) from premalignant leukoplakia or squamous cell carcinoma. Burning mouth syndrome often involves the tongue and has responded to treatment with alpha-lipoic acid, clonazepam, and cognitive behavior therapy in controlled trials. Several trials have also confirmed the effectiveness of surgical division of tongue-tie (ankyloglossia), in the context of optimizing the success of breastfeeding compared with education alone. Tongue lesions of unclear etiology may require biopsy or referral to an oral and maxillofacial surgeon, head and neck surgeon, or a dentist experienced in oral pathology.

Novel intraoral phenotypes in hyperimmunoglobulin-E syndrome.

AIM: Hyperimmunoglobulin-E syndrome (HIES) is a primary immunodeficiency characterized by eczema, recurrent skin and lung infections with pneumatocoele formation, and extremely elevated serum immunoglobulin-E. The precise immunologic defect and genetic etiology remain unknown. Non-immunologic findings include characteristic facial features (prominent forehead, fleshy nasal tip, and increased interalar distance); skeletal involvement (pathological fractures, scoliosis, and craniosynostosis); and retention of primary teeth. This study aims to characterize intraoral soft tissue findings in HIES patients. METHODS: Sixty HIES patients (4-54 years, 27 males, 33 females) received intraoral and radiographic evaluations. Chronological dental development was also assessed. RESULTS: Lesions of the hard palate and dorsal tongue were found in 55% and 60% of patients, respectively. Palatal lesions ranged from a generalized surface keratosis to a midline sagittal fibrotic bridge. Tongue lesions consisted of multiple fissures and a midline cleft. On the lip and buccal mucosa, keratotic plaques and/or surface fissures were found in 8% and 23% of patients, respectively. Manifested in 76.7% of patients, the intraoral lesions were significantly more prevalent than the characteristic facial traits (P=0.0013). CONCLUSIONS: Alterations in oral mucosa and gingiva were present in the majority of HIES patients. These novel intraoral findings may facilitate the diagnosis of HIES.

Traumatic ulcerative granuloma with stromal eosinophilia of the oral mucosa: histological and immunohistochemical analysis of three cases.

Traumatic ulcerative granuloma with stromal eosinophilia is an ulcerated oral lesion which pathogenesis is still unclear. Clinically, this disease is characterized by the presence of chronic ulcerative lesions with elevated and indurated borders in the oral mucosa. It usually develops rapidly and persists for several weeks or months, it presents mainly on the tongue but other areas such as gingival, cheek and vestibular mucosa may also be involved. We report three cases showing their clinical, histological and immunohistochemical analysis. In this study the authors underline the importance of a correct differential diagnosis and monitoring of these patients in order to prevent the development of possible serious complications.

Repeated massive tongue swelling due to the combined use of estramustine phosphate and angiotensin-converting enzyme inhibitor.

A 70-year-old man presenting with a chief complaint of tongue swelling had been diagnosed with prostate cancer 1 year earlier. He had been on an oral angiotensin-converting enzyme inhibitor (ACE) inhibitor for hypertension for 20 years. Two months before the first of 4 episodes of tongue swelling within a period of 40 days, he had been prescribed oral estramustine phosphate (EMP) for the prostate cancer. He was admitted to our hospital for the evaluation after massive swelling of the tongue and epiglottis which necessitated tracheotomy. Food allergies, allergic reactions to environmental factors, and hereditary angioneurotic edema were excluded. Massive swelling of the tongue and epiglottis disappeared completely after EMP was discontinued. We concluded that angioedema was induced by EMP used concurrently with the ACE inhibitor.

Endodontic treatment in cases of allergic reaction to rubber dam.

The prevalence and severity of latex allergies have rapidly increased recently. This article presents two cases of patients with rubber latex allergy. The patient in case A was unaware of her sensitivity to latex and presented symptoms of contact dermatitis-stomatitis during endodontic treatment. The patient in case B reported latex allergy before the initiation of the treatment and a different approach was followed. Certain aspects of latex allergy related to the endodontic treatment are discussed. Moreover, a protocol is proposed for treatment of patients with latex hypersensitivity with safety.

Tongue lesions: what can it be?

In the course of the examination of a patient their tongue is easily visualized and with the use of gauze can be gently dried, held with gauze and stretched for inspection of all aspects. This can be followed by palpation of both the tongue and the floor of mouth. In this manner, any altered tissue is likely to be detected and, with or without further investigations, a diagnosis established. Appropriate management can then be undertaken. A wide range of conditions may involve the tongue and this article outlines some of these using a classification based on aetiology.

Mucosal cell-mediated immunological changes associated with experimental graft-versus-host disease.

This study examined the histological changes and local cellular immune response induced within the lingual mucosa in an allogeneic F1 hybrid rat model of graft-versus-host disease (GvHD) with a view to studying oral lymphocyte-epithelial cell reactions. Highest levels of disease, as reflected by both a GvHD index and the extent of the oral mucosal changes, were obtained using primed donor (Lewis rats) splenocytes and irradiated hosts (Lew/Da rats). The lingual mucosae of test animals were characterised by irregular epithelial keratosis, an absence of basal cell liquefaction and a diffuse inflammatory cell infiltrate, histological features consistent with an oral lichenoid tissue reaction. Immunohistochemical studies showed that mucosal involvement was characterised by infiltration of the lamina propria by NK cells (CD8+, CD5-), "activated" cells (CD25+) and T cells (CD5+) with selective migration of the latter, including a CD5+, CD8- subset (helper/inducer T cell), into the epithelium. Epithelial expression of Ia was invariably associated with these inflammatory cell infiltrates and correlated with the GvHD index. These findings suggest the presence of local mucosal T cell activation in the absence of detectable epithelial cell damage, which may be equivalent to the early initiating events in the pathogenesis of oral lichen planus. However, whilst experimental graft-versus-host disease appears to be a useful model for studying lymphocyte-epithelial interactions, the induced oral mucosal changes are more consistent with a lichenoid reaction rather than lichen planus.

Tuberculosis of the tongue. A case report with immunologic study.

A case of tuberculosis of the tongue in a 59-year-old woman with active pulmonary tuberculosis is described. The lingual tuberculosis was considered to be a secondary infection from the pulmonary disease, but the oral lesions were, in fact, noticed prior to recognition of the pulmonary lesion. This case was marked by a multiplicity or oral lesions arising on the bilateral surfaces of the tongue. Immunologic investigation revealed that cell-mediated immune responses in the patient were within the normal range in terms of the PPD skin test, DNCB skin test, lymphocyte transformation test, and subpopulation of the peripheral blood lymphocytes. Rosette-forming assay on the frozen sections disclosed that T-lymphocytes and macrophages were predominant in the lymphoid cells infiltrating the tuberculous lesion.