Nasopalatine canal cyst, a diagnostic twist.

We present a case of nasopalatine duct cyst in a 35-yearold female. The cyst was diagnosed based on the presence of only one clinical symptom and no obvious clinical signs, which is a relatively rare occurrence. However, the radiographic and histological presentation of this lesion was typical of a nasopalatine duct cyst. Therefore, this case report aims to highlight the variable presentations of the nasopalatine cyst, which is often misdiagnosed and treated as an endodontic infection.

Long term outcome of chondromesenchymal hamartoma of sinonasal cavity: a rare entity.

Nasal chondromesenchymal hamartoma in the sinonasal cavity is an unusual entity mostly found in young infants and children. We present the case of a nasal chondromesenchymal hamartoma in a young female. NCMH has a favourable outcome that shows neither recurrence nor any malignant behaviour.

Interferon inducible GBPs restrict Burkholderia thailandensis motility induced cell-cell fusion.

Innate immunity responds to pathogens by producing alarm signals and activating pathways that make host cells inhospitable for pathogen replication. The intracellular bacterium Burkholderia thailandensis invades the cytosol, hijacks host actin, and induces cell fusion to spread to adjacent cells, forming multinucleated giant cells (MNGCs) which promote bacterial replication. We show that type I interferon (IFN) restricts macrophage MNGC formation during B. thailandensis infection. Guanylate-binding proteins (GBPs) expressed downstream of type I IFN were required to restrict MNGC formation through inhibition of bacterial Arp2/3-dependent actin motility during infection. GTPase activity and the CAAX prenylation domain were required for GBP2 recruitment to B. thailandensis, which restricted bacterial actin polymerization required for MNGC formation. Consistent with the effects in in vitro macrophages, Gbp2-/-, Gbp5-/-, GbpChr3-KO mice were more susceptible to intranasal infection with B. thailandensis than wildtype mice. Our findings reveal that IFN and GBPs play a critical role in restricting cell-cell fusion and bacteria-induced pathology during infection.

Chondrodermatitis Nodularis Nasi: A Case Report of a Rare Variant of Chondrodermatitis Nodularis Helicis.

ABSTRACT: Chondrodermatitis nodularis helicis is an inflammatory condition affecting the helix or antihelix of the ear. It is commonly described as a solitary, painful, ulcerated nodule affecting the cartilage or skin because of continuous pressure, trauma, sun exposure, or ischemic changes. In this case report, we present a rare variant of chondrodermatitis, named chondrodermatitis nodularis nasi, which affects the skin and cartilage of the nose. Clinical resemblance to neoplasms of the skin such as squamous cell carcinoma and basal cell carcinoma requires that a biopsy be performed for definitive diagnosis. On histopathological examination, chondrodermatitis presents as a central ulcer bordered by epidermal hyperplasia, hypergranulosis, hyperkeratosis, and parakeratosis with fibrin deposits in the papillary dermis admixed with sparse inflammatory cells. Included in this case report is a discussion of the clinical and histopathology of chondrodermatitis nodularis nasi. With increased frequency of diagnosing this rare variant, better management and treatments can be explored.

Eosinophilic Angiocentric Fibrosis of the Nasal Cavities: A Report of an Uncommon Lesion with Emphasis on the Etiology and Differential Diagnosis.

Background and Objectives: Eosinophilic angiocentric fibrosis (EAF) is an indolent but sometimes locally destructive lesion with a predilection for the sinonasal tract. Although it was first described in 1983, its etiology remains unknown. Some authors initially attributed EAF to trauma, hypersensitivity, and/or surgical manipulation, while it has been recently suggested to include EAF within the spectrum of IgG4-related systemic diseases. Materials and Methods: We report an uncommon case of idiopathic EAF in a 76-year-old male who developed two bilateral tumefactive masses in the nasal cavities. Results: As the histological examination showed a subepithelial proliferation of fibroblasts along with sclero-hyaline fibrosis around small-sized vessels (an "onion skin-like" pattern) and an eosinophils-rich inflammatory infiltrate, a diagnosis of EAF was rendered. The differential diagnosis included granuloma faciale, Wegener's granulomatosis, and Churg-Strauss syndrome. Conclusions: Pathologists should be aware of the possibility that this lesion can be part of the wide spectrum of IgG4-related systemic diseases by performing IgG4 investigations to assess adherence to IgG4-related systemic disease criteria.

Rosai-Dorfman-Destombes (RDD) disease presenting as palindromic rheumatism.

BACKGROUND: Rosai-Dorfman-Destombes (RDD) disease, is a rare proliferative and inflammatory disorder of non-Langerhans cell histiocytes. CASE PRESENTATION: We report a 35-year-old woman, who originally presented with recurrent episodes of lower extremity joint/bone pain and chronic nasal stuffiness and congestion. Her worsening nasal congestion was due to an obstructing nasal cavity lesion which was subsequently biopsied. Pathology was consistent with RDD. 18F-FDG PET images demonstrated intense uptake in the paranasal sinuses and a large pelvic lymph node. Focal osseous lesions with intense 18F-FDG uptake were also observed in the lower extremity, corresponding to areas of peri-articular pain. Rheumatologic work-up was consistent with palindromic rheumatism. She was diagnosed with immune-related disseminated RDD, presenting as palindromic rheumatism. CONCLUSIONS: This is the first case of RDD presenting as palindromic rheumatism. RDD should be considered as a possible but rare diagnosis in young patients with sinus-related symptoms and lymphadenopathy. The disease can on rare occasions be disseminated and can also present as immune-related RDD, such as in this patient.

Sarcoidosis of the ear, nose and throat: A review of the literature.

OBJECTIVES: Sarcoidosis is a multisystemic inflammatory disease with extrathoracic manifestations, most commonly affecting the young and middle-aged, female and Black populations. Diagnosis usually requires evidence of non-caseating granulomata and, when treated, prognosis is usually favourable. We aim to establish the incidence, clinical features and optimal treatment of ENT manifestations of this disease. DESIGN: We performed a PubMed literature review to determine the evidence base supporting this. RESULTS: ENT manifestations are present in 5%-15% of patients with sarcoidosis, often as a presenting feature, and require vigilance for swift recognition and coordinated additional treatment specific to the organ. Laryngeal sarcoidosis presents with difficulty in breathing, dysphonia and cough, and may be treated by speech and language therapy (SLT) or intralesional injection, dilatation or tissue reduction. Nasal disease presents with crusting, rhinitis, nasal obstruction and anosmia, usually without sinus involvement. It is treated by topical nasal or intralesional treatments but may also require endoscopic sinus surgery, laser treatment or even nasal reconstruction. Otological disease is uncommon but includes audiovestibular symptoms, both sensorineural and conductive hearing loss, and skin lesions. CONCLUSIONS: The consequences of ENT manifestations of sarcoidosis can be uncomfortable, disabling and even life-threatening. Effective management strategies require good diagnostic skills and use of specific therapies combined with established treatments such as corticosteroids. Comparisons of treatment outcomes are needed to establish best practice in this area.

Black Fungus Complicated with COVID-19 in a Man with Underlying Non-Hodgkin's Lymphoma.

COVID-19 is a disease reported to suppress cellular immunity. This may lead to the development of opportunistic infections, among others black fungus, or mucormycosis. On the other hand, pre-existing defect in immunity may render patients susceptible to both mucormycosis and COVID-19. Mucormycosis is a relatively rare fungal infection with rapid progression unless diagnosed promptly and treated adequately, and urgent surgical and medical intervention is lifesaving. The manifestation of mucormycosis largely depends on the presence of exposure to the pathogen and the existing risk factor of the host. As black fungus is locally invasive, the majority of cases will involve tissue damage with local destruction and contiguous spread to nearby structure. We here with present a case of black fungus complicated with COVID-19 in a man with underlying non-Hodgkin's lymphoma.

Chondrodermatitis nodularis nasi.

Chondrodermatitis nodularis helicis is a relatively common ulcerative, inflammatory condition affecting the skin, and cartilage of the ear. We present a case of chondrodermatitis affecting the skin and cartilage of the nose, designated chondrodermatitis nodularis nasi. Clinically, chondrodermatitis presents as a singular, painful, ulcerated papule or nodule that results from prolonged pressure, trauma, sun exposure, or vascular compromise. The clinical features resemble those seen in skin cancer, and a biopsy is often required to secure the diagnosis. Histopathologic analysis of chondrodermatitis demonstrates a central ulcer, beneath which there is fibrinoid necrosis of the dermis and inflammatory infiltrates. The ulcer is bordered by pseudoepitheliomatous hyperplasia with underlying granulation tissue. In addition to a clinical and morphologic description of chondrodermatitis nodularis nasi, a discussion of etiological factors and differential diagnoses is included. Increased recognition and reporting of the condition will allow for the exploration of optimal treatment strategies.

A Very Rare Localization of Rhinolith: Fossa of Rosenmuller.

Sixty-five-year old male patient applied to our clinic with nonspecific complaints such as tinnitus, otalgia, headache, halitosis, and persistent globus pharyngeus which had been continued for 5 years. His complaints persisted despite different extensive treatments. In nasoendoscopy, a yellow-colored mass with purulent discharge around was observed in eustachian tube opening in left fossa of Rosenmüller. Computed tomography scan showed a mass of 6 × 6 mm size in left fossa of Rosenmüller. After excision of the mass under local anesthesia, significant reductions were observed in patient's symptoms. Histopathology of the mass was reported as rhinolith. Although fossa of Rosenmüller is a quite rare localization for rhinoliths, it needs to be considered in differential diagnosis of patients with nonspecific symptoms such as halitosis, globus pharyngeus, and otalgia. This report represents a patient with rhinolith localized in fossa of Rosenmüller as the 2nd report in the literature.

Nasal eosinophilic angiocentric fibrosis with IgG4-positive plasma cell infiltration.

INTRODUCTION: Eosinophilic angiocentric fibrosis (EAF) is a rare lesion that predominantly affects the upper respiratory tract. Its etiology is unknown and it has been recently associated with the IgG4- related disease (IgG4-RD) spectrum. To the author's knowledge, this is the sixth case report of the relationship between EAF and IgG4-RD. CASE REPORT: We report the case of a 37-year-old woman with nasal deformity and facial pain. The lesion was surgically excised. Histological examination revealed features of EAF with an IgG4/IgG plasma cell ratio ≷73% and 31 IgG4 stained cells per high power field. No clinical or radiological recurrence was detected during follow-up. Serum IgG4 quantification one year after surgery was within normal limits. DISCUSSION: The relationship between both entities may have therapeutic impact because IgG4-RD of the head and neck has a high remission rate with corticosteroids and immunosuppressive therapy. Additional reports of this infrequent disease are necessary to elucidate appropriate treatment and prognosis.

Herpes simplex virus of the nose masquerading as invasive fungal sinusitis: A pediatric case series.

The management of invasive fungal sinusitis differs greatly from the management of herpes simplex virus (HSV) of the nose in immunocompromised patients. However, the diagnosis may be uncertain and a delay in treatment can lead to mortality. Here we describe the successful medical management of a series of immunocompromised pediatric patients with HSV lesions of the nose with the initial concern for invasive fungal sinusitis. The diagnosis of HSV herpes was supported by positive polymerase chain reaction (PCR) testing of the nasal lesion. To our knowledge, these are the first cases described in the pediatric literature, emphasizing the need to include this entity on the differential.

Giant isolated transcranial Rosai-Dorfman disease with diffuse involvement of nasal and paranasal tissues: case report and literature review.

Rosai-Dorfman disease (RDD) is an uncommon systemic histioproliferative disease process characterized by sinus histiocytosis with massive lymphadenopathy, and isolated transcranial RDD (ITRDD) is extremely rare. We report a patient with giant ITRDD with diffuse involvement of nasal and paranasal tissues, showing favorable response to postoperative steroid therapy.

Unusual Case of Combined Gliomeningeal Heterotopia on the Nose of an Infant.

Nasal glial heterotopia ("nasal glioma") and cutaneous heterotopic meningeal nodules ("primary cutaneous meningioma") are rare congenital lesions characterized by the presence of heterotopic mature cerebral tissues. Nasal glial heterotopia occurs predominantly in the nasal area and typically does not contain meningothelial elements, whereas heterotopic meningeal nodules occur predominantly on the scalp and do not contain glial elements. In this article, we report an unusual case of cutaneous heterotopia on the nose of an infant composed of both glial and meningothelial elements. The glial component was characterized by irregular islands of predominantly astrocytic cells, on a fibrillary background. The meningothelial component was characterized by bland ovoid cells with focal intranuclear inclusions forming whorled arrangements, with associated psammomatous calcification. To our knowledge, this is the first time such a lesion has been documented. It has also provided us with an opportunity to review the literature regarding heterotopic deposits of both glial and meningothelial tissues.

Generation of Th17 cells in response to intranasal infection requires TGF-ß1 from dendritic cells and IL-6 from CD301b+ dendritic cells.

Intranasal (i.n.) infections preferentially generate Th17 cells. We explored the basis for this anatomic preference by tracking polyclonal CD4(+) T cells specific for an MHC class II-bound peptide from the mucosal pathogen Streptococcus pyogenes. S. pyogenes MHC class II-bound peptide-specific CD4(+) T cells were first activated in the cervical lymph nodes following i.n. inoculation and then differentiated into Th17 cells. S. pyogenes-induced Th17 formation depended on TGF-ß1 from dendritic cells and IL-6 from a CD301b(+) dendritic cell subset located in the cervical lymph nodes but not the spleen. Thus, the tendency of i.n. infection to induce Th17 cells is related to cytokine production by specialized dendritic cells that drain this site.

Recapitulation of Unilateral Cleft Lip Nasal Deformity on Normal Nasal Structure: A Finite Element Model Analysis.

Cleft lip nasal deformity has been challenging to plastic surgeons. A better understanding of the biomechanical aspect of the cleft nose would contribute to a better correction. In this study, finite element model of a normal nose was constructed and loaded with forces to recapitulate the unilateral cleft lip nasal deformity. Tether at the alar base was simulated by a laterally directed force at the lateral crus, and tether at the columella base by a posteriorly directed force at the medial crus. The equivalent von-Mises stress and the total deformation consequent to different patterns of loading were captured. In accordance with clinical observations, unilaterally loaded forces caused deformation on both sides of the nose. A correlation between the patterns of loading and different cleft lip nasal deformities was documented in detail. When set at the same force magnitude, tether at the columella base led to more extensive changes in the nasal morphology and higher level of stress than at the alar base. Clear identification of major pathological tethers in the nasolabial region might lead to more accurate and stable correction of cleft lip nasal deformities.

Evaluation of the Characteristics and Association Between Schneiderian Membrane Thickness and Nasal Septum Deviation.

OBJECTIVE: Nasal septum deviation (NSD) is an anomaly that disturbs normal mucus drainage in sinuses, leading to recurrent sinusitis. Sinusitis, infections, and mucosal irritations are the potential causes of Schneiderian membrane thickening (SMT). The aim of this study was to evaluate the characteristics and association between NSD and SMT along with factors such as age, gender, and tooth groups using cone-beam computed tomography (CBCT) imaging. METHODS: The maxillary sinus and nasal cavity of 249 patients (136 females and 113 males) were examined retrospectively using CBCT. The statistical analysis was performed to determine the association between SMT, NSD, gender, and age. RESULTS: No statistically significant relation was detected between NSD and SMT. The NSD was found in 50.6% of patients without gender predilection. Age was not associated with the presence of NSD. The mean thickness of the membrane was least in the third molar region and highest in the first molar region. The average SMT was found to be higher in males for all the examined tooth areas (P ≤ 0.05). CONCLUSION: A retrospective evaluation of CBCT images emphasized the concept that NSD was not a factor that affected the thickening of maxillary sinus mucosa. The CBCT imaging could be used for evaluating the maxillary sinuses and nasal septum.

The Effect of Unilateral Concha Bullosa on Olfactory Bulb Volume: an Assessment by Magnetic Resonance Imaging.

BACKGROUND: In this study, the authors compared the right and left olfactory bulb volumes by magnetic resonance image findings of patients with unilateral concha bullosa (CB). METHODS: The cranial magnetic resonance imaging studies of 24 patients having unilateral CB were reviewed. There were 10 males and 14 females ranging in age from 29 to 51 years (mean age, 29 ±â€Š15.2 years). The volumes of both olfactory bulbs (contralateral and ipsilateral to the concha bullosa side) were calculated by using the computer program. RESULTS: The average values for olfactory bulb volumes were 46.57 ±â€Š8.03 mm in the CB side of the nasal cavity and 54.80 ±â€Š10.031 mm in the normal side of the nasal cavity. We found a statistically significant difference in olfactory bulb volume between the contralateral and ipsilateral to the concha bullosa side of the patients (t = -3.08 and P < 0.01 for CB side, t = -3.47 and P < 0.01 for left normal side). CONCLUSION: Our results may point out a possible relationship between CB and ipsilateral decreased olfactory bulb volume, yet the exact mechanism still remains unclear. Decreased nasal air flow on the CB side may be attributed to the pathophysiologic mechanism of this finding. Also, the significant reduction in olfactory bulb volumes ipsilateral to CB compared with the contralateral olfactory bulb volumes seems to support our hypothesis.

Nasal Cavity Actinomycosis Mimicking Rhinolith.

Actinomycosis is a subacute-to-chronic bacterial infection caused by filamentous, gram-positive, non-acid-fast, anaerobic-to-microaerophilic bacteria. It is characterized by continuous spread, suppurative and granulomatous inflammation, and formation of multiple abscesses and sinus tracts that may discharge sulfur granules. Although the most common clinical forms of actinomycosis are cervicofacial, involvement of the nose and paranasal sinuses are extremely rare. Furthermore, nasal cavity actinomycosis mimicking rhinolith has not been reported in the literature previously. This article reports a patient of nasal cavity actinomycosis causing symptoms similar to those of a rhinolith, which was successfully treated by surgical debridement and antibiotic therapy.