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1.
Laryngoscope ; 131(8): 1816-1820, 2021 08.
Article in English | MEDLINE | ID: mdl-32902896

ABSTRACT

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.


Subject(s)
Autoimmune Diseases/pathology , Laryngostenosis/immunology , Tongue Diseases/immunology , Tongue/immunology , Adult , Aged , Autoimmune Diseases/immunology , Chi-Square Distribution , Constriction, Pathologic/immunology , Constriction, Pathologic/pathology , Disease Progression , Female , Granulomatosis with Polyangiitis/immunology , Granulomatosis with Polyangiitis/pathology , Humans , Laryngostenosis/pathology , Male , Middle Aged , Odds Ratio , Retrospective Studies , Tongue/pathology , Tongue Diseases/pathology
2.
BMC Complement Altern Med ; 17(1): 313, 2017 Jun 14.
Article in English | MEDLINE | ID: mdl-28615025

ABSTRACT

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.


Subject(s)
Anti-Inflammatory Agents/administration & dosage , Fabaceae/chemistry , Plant Extracts/administration & dosage , Plant Oils/administration & dosage , Resins, Plant/administration & dosage , Tongue Diseases/drug therapy , Animals , Humans , Male , Rats , Rats, Wistar , Tongue Diseases/immunology , Tongue Diseases/physiopathology , Wound Healing
3.
Transplantation ; 101(6): 1441-1448, 2017 06.
Article in English | MEDLINE | ID: mdl-27336393

ABSTRACT

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.


Subject(s)
Inflammation/immunology , Inflammation/pathology , Organ Transplantation/adverse effects , Tongue Diseases/immunology , Tongue Diseases/pathology , Adolescent , Adult , Age Factors , Aged, 80 and over , Biomarkers/analysis , Biopsy , Brazil , Child , Female , Humans , Immunohistochemistry , Immunosuppressive Agents/administration & dosage , Male , Middle Aged , Sweden , Treatment Outcome
4.
Hum Pathol ; 59: 147-151, 2017 01.
Article in English | MEDLINE | ID: mdl-27569297

ABSTRACT

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.


Subject(s)
Colitis, Ulcerative/virology , Cytomegalovirus Infections/virology , Epstein-Barr Virus Infections/virology , Hematopoietic Stem Cell Transplantation/adverse effects , Herpesvirus 4, Human/isolation & purification , Lymphoma, Large B-Cell, Diffuse/surgery , Lymphoproliferative Disorders/virology , Opportunistic Infections/virology , Tongue Diseases/virology , Ulcer/virology , Biopsy , Colitis, Ulcerative/diagnosis , Colitis, Ulcerative/immunology , Cytomegalovirus Infections/diagnosis , Cytomegalovirus Infections/immunology , Disease Progression , Epstein-Barr Virus Infections/diagnosis , Epstein-Barr Virus Infections/drug therapy , Epstein-Barr Virus Infections/immunology , Fatal Outcome , Female , Herpesvirus 4, Human/genetics , Humans , Immunocompromised Host , Immunohistochemistry , Immunosuppressive Agents/adverse effects , In Situ Hybridization , Lymphoma, Large B-Cell, Diffuse/diagnosis , Lymphoproliferative Disorders/diagnosis , Lymphoproliferative Disorders/drug therapy , Lymphoproliferative Disorders/immunology , Middle Aged , Opportunistic Infections/diagnosis , Opportunistic Infections/immunology , Polymerase Chain Reaction , RNA, Viral/genetics , Recurrence , Time Factors , Tongue Diseases/diagnosis , Tongue Diseases/immunology , Ulcer/diagnosis , Ulcer/immunology
6.
Dermatol Online J ; 21(3)2014 Dec 14.
Article in English | MEDLINE | ID: mdl-25780970

ABSTRACT

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.


Subject(s)
Histoplasmosis/diagnosis , Tongue Diseases/diagnosis , Antifungal Agents/therapeutic use , Female , Histoplasmosis/drug therapy , Histoplasmosis/immunology , Humans , Immunocompetence , Itraconazole/therapeutic use , Middle Aged , Tongue Diseases/drug therapy , Tongue Diseases/immunology , Tongue Diseases/microbiology
8.
Article in English | MEDLINE | ID: mdl-22901649

ABSTRACT

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.


Subject(s)
Autoimmune Diseases/diagnosis , Autoimmune Diseases/immunology , Immunoglobulin G/immunology , Sclerosis/diagnosis , Sclerosis/immunology , Tongue Diseases/diagnosis , Tongue Diseases/immunology , Autoimmune Diseases/drug therapy , Biopsy , Carcinoma, Squamous Cell/diagnosis , Diagnosis, Differential , Diagnostic Imaging , Female , Humans , Immunohistochemistry , Middle Aged , Mouth Neoplasms/diagnosis , Sclerosis/drug therapy , Steroids/therapeutic use , Tongue Diseases/drug therapy
9.
Oral Maxillofac Surg ; 15(3): 179-82, 2011 Sep.
Article in English | MEDLINE | ID: mdl-20625784

ABSTRACT

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.


Subject(s)
Eosinophilia/psychology , Oral Ulcer/psychology , Stress, Psychological/immunology , Tongue Diseases/psychology , Abdominal Pain/immunology , Abdominal Pain/psychology , Adult , Carcinoma, Squamous Cell/diagnosis , Diagnosis, Differential , Eosinophilia/immunology , Follow-Up Studies , Humans , Male , Oral Ulcer/immunology , Psychoneuroimmunology , Tongue Diseases/immunology , Tongue Neoplasms/diagnosis
10.
Am Fam Physician ; 81(5): 627-34, 2010 Mar 01.
Article in English | MEDLINE | ID: mdl-20187599

ABSTRACT

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.


Subject(s)
Tongue Diseases/diagnosis , Atrophy , Carcinoma, Squamous Cell/diagnosis , Glossitis/microbiology , Glossitis/pathology , Humans , Leukoplakia, Hairy/diagnosis , Leukoplakia, Hairy/etiology , Lichen Planus, Oral/diagnosis , Lichen Planus, Oral/immunology , Nutrition Disorders/complications , Primary Health Care , Tongue Diseases/epidemiology , Tongue Diseases/etiology , Tongue Diseases/immunology , Tongue Neoplasms/diagnosis
11.
Oral Dis ; 14(1): 73-81, 2008 Jan.
Article in English | MEDLINE | ID: mdl-18173452

ABSTRACT

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.


Subject(s)
Hypergammaglobulinemia/immunology , Immunoglobulin E/immunology , Mouth Diseases/immunology , Adolescent , Adult , Child , Child, Preschool , Female , Fibrosis , Humans , Leukoplakia, Oral/immunology , Lip Diseases/immunology , Male , Middle Aged , Mouth Mucosa/immunology , Odontogenesis/immunology , Palate, Hard/immunology , Phenotype , Syndrome , Tongue/abnormalities , Tongue Diseases/immunology
12.
Minerva Stomatol ; 56(1-2): 73-9, 2007.
Article in English, Spanish | MEDLINE | ID: mdl-17287709

ABSTRACT

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.


Subject(s)
Eosinophilic Granuloma/pathology , Lymphoproliferative Disorders/pathology , Oral Ulcer/pathology , Tongue Diseases/pathology , Adolescent , Adult , CD3 Complex/analysis , CD5 Antigens/analysis , Carcinoma, Squamous Cell/diagnosis , Child , Diagnosis, Differential , Eosinophilic Granuloma/classification , Eosinophilic Granuloma/diagnosis , Eosinophilic Granuloma/immunology , Female , Humans , Ki-1 Antigen/analysis , Lymphoproliferative Disorders/classification , Lymphoproliferative Disorders/immunology , Male , Oral Ulcer/classification , Oral Ulcer/diagnosis , Oral Ulcer/immunology , Remission, Spontaneous , Stromal Cells/pathology , T-Lymphocyte Subsets/chemistry , T-Lymphocyte Subsets/pathology , Tongue Diseases/diagnosis , Tongue Diseases/immunology , Tongue Neoplasms/diagnosis
13.
J Investig Allergol Clin Immunol ; 16(6): 388-90, 2006.
Article in English | MEDLINE | ID: mdl-17153888

ABSTRACT

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.


Subject(s)
Angioedema/chemically induced , Angiotensin-Converting Enzyme Inhibitors/adverse effects , Antineoplastic Agents, Hormonal/adverse effects , Drug Hypersensitivity/therapy , Estramustine/adverse effects , Aged , Angiotensin-Converting Enzyme Inhibitors/immunology , Antineoplastic Agents, Hormonal/immunology , Estramustine/immunology , Humans , Male , Tongue Diseases/chemically induced , Tongue Diseases/immunology , Tracheotomy
14.
Oral Microbiol Immunol ; 21(4): 223-30, 2006 Aug.
Article in English | MEDLINE | ID: mdl-16842506

ABSTRACT

INTRODUCTION: Neutrophils are the main opponents of Candida albicans in chronic hyperplastic candidosis. They migrate from the circulation to the epithelium where they form microabscesses. We therefore hypothesized that the neutrophil chemokine interleukin-8 (IL-8) might play a role in the neutrophil-Candida interaction. METHODS: Biopsies from patients with chronic hyperplastic candidosis (n = 10) were stained using the avidin-biotin-peroxidase complex protocol for IL-8 and IL-8 receptor A and were compared to healthy control mucosa (n = 3). A set of C. albicans agar sections was similarly analysed. RESULTS: In chronic hyperplastic candidosis lesions IL-8 was strongly expressed in both vascular endothelium and mucosal epithelium. Many resident and immigrant inflammatory cells, including intraepithelial neutrophils, were IL-8 receptor A positive. In addition, IL-8 (or an analogue) was found in the candidal mother cell in chronic hyperplastic candidosis and in agar, whereas the tips of the hyphae expressed IL-8 receptor A (or an analogue). CONCLUSION: IL-8 may play a role in the recruitment of neutrophils from the vascular compartment to the epithelial microabscesses. C. albicans may have developed an ability to sense IL-8. The IL-8 ligand-receptor interaction may help to direct the growth of the IL-8-receptor-containing tips of the hyphae away from the IL-8-producing candidal cell body (a centrifugal growth pattern to facilitate host tissue penetration). Later, this ability might help to keep the vulnerable hyphal tips away from areas with high concentrations of host IL-8 and candidacidal neutrophils. We suggest that this phenomenon, in contrast to chemotropism, is named chemophobia.


Subject(s)
Candidiasis, Oral/immunology , Interleukin-8/analysis , Receptors, Interleukin-8A/analysis , Adult , Aged , Aged, 80 and over , Candida albicans/immunology , Chemotaxis, Leukocyte/immunology , Chronic Disease , Endothelium, Vascular/immunology , Epithelium/immunology , Female , Humans , Hyperplasia , Immunoenzyme Techniques , Interleukin-8/genetics , Male , Middle Aged , Mouth Mucosa/immunology , Mouth Mucosa/microbiology , Neutrophil Infiltration/immunology , Neutrophils/immunology , Palate/immunology , Palate/microbiology , Receptors, Interleukin-8A/genetics , Tongue Diseases/immunology , Tongue Diseases/microbiology
17.
J Endod ; 28(11): 787-9, 2002 Nov.
Article in English | MEDLINE | ID: mdl-12470026

ABSTRACT

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.


Subject(s)
Latex Hypersensitivity/immunology , Root Canal Therapy/instrumentation , Rubber Dams/adverse effects , Aged , Burning Mouth Syndrome/immunology , Clinical Protocols , Equipment Design , Female , Gloves, Surgical , Humans , Lip Diseases/immunology , Polyvinyl Chloride , Safety , Stomatitis/immunology , Tongue Diseases/immunology , Vinyl Compounds
18.
Ann R Australas Coll Dent Surg ; 15: 106-10, 2000 Oct.
Article in English | MEDLINE | ID: mdl-11709914

ABSTRACT

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.


Subject(s)
Tongue Diseases/diagnosis , Diagnosis, Differential , Humans , Mouth Floor/pathology , Palpation , Tongue/abnormalities , Tongue/injuries , Tongue Diseases/etiology , Tongue Diseases/immunology , Tongue Diseases/microbiology , Tongue Neoplasms/diagnosis
19.
HNO ; 47(12): 1063-8, 1999 Dec.
Article in German | MEDLINE | ID: mdl-10654184

ABSTRACT

Hyperimmunoglobulin E syndrome (Hiob syndrome or Buckley syndrome) is a rare disorder of the immune system that can show characteristic manifestations in the head and neck. Typical symptoms are fever, recurrent urticarial rashes, lymphadenitis, and bacterial infections of the skin and various parenchymatous organs. Diagnosis is established by elevated serum IgE concentrations with the absence of any signs of allergy or parasitic disease. We present our clinical experiences in managing of a 29-year old woman whose hyper IgE syndrome was diagnosed initially during of the treatment of lymphatic hyperplasia of the base of the tongue although she had typical symptoms of hyper IgE syndrome for some years. High-dose intravenous immunoglobulin therapy (IVIG) was found to be well-tolerated and effective. More than one year after a single course of immunoglobin therapy symptoms markedly improved. Current knowledge and therapeutic options in hyper IgE syndrome are discussed. We recommend that IVIG be considered as one of the first choices in the treatment of hyper IgE syndrome.


Subject(s)
Immunoglobulins, Intravenous/administration & dosage , Job Syndrome/therapy , Otorhinolaryngologic Diseases/therapy , Tongue Diseases/therapy , Adult , Diagnosis, Differential , Female , Humans , Hyperplasia , Job Syndrome/immunology , Otorhinolaryngologic Diseases/immunology , Tongue/pathology , Tongue Diseases/immunology
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