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      A rare case of sarcoidosis involving the middle turbinates: an incidental diagnosis

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      1 , , 1 , 2 , 1
      Diagnostic Pathology
      BioMed Central

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          Abstract

          Background

          Sarcoidosis is a chronic, systemic granulomatous disease of unknown etiology that features noncaseating granulomas in many body regions. Sinonasal involvement is rare but is also suspected to be underreported.

          Case presentation

          We present the case of a 39-year-old woman who was incidentally diagnosed with isolated sarcoidosis involving the middle turbinates. Histopathologic examination of resected concha bullosa material and an extensive panel of diagnostic tests revealed a diagnosis of isolated sarcoidosis. Since no systemic manifestations were detected, topical corticosteroid (nasal spray) was administered in the postoperative period. Throughout the 12 months after surgery, the patient remained free of symptoms and all nasal endoscopy examinations were normal.

          Conclusion

          Although isolated nasal involvement of sarcoidosis is rare, otorhinolaryngologists should consider this condition in a differential diagnosis for sinonasal complaints.

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          Most cited references15

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          Sinonasal sarcoidosis: review and report of fifteen cases.

          Sinonasal sarcoidosis remains a poorly understood and uncommon chronic granulomatous disease of unclear origin. We have attempted to characterize the main clinical and radiologic criteria for diagnosis and to discuss the treatment. A retrospective study of 15 cases of chronic, symptomatic, and biopsy-proven sinonasal sarcoidosis and a review of the literature are realized. Among the 15 patients, there were 8 women and 7 men with a mean age of 44 years. The most frequent presentation was a chronic, often crusty, rarely destructive inflammatory rhinosinusitis with nodules on the septum and/or the turbinates. Pulmonary sarcoidosis was associated in 12 cases. Involvement of the nasopharynx, the pharyngolarynx, the skin, the lachrymal and salivary glands, and the liver was associated in some cases. Levels of angiotensin-converting enzyme were elevated in 10 cases and normal in 3 cases. Gallium scan performed in three cases was positive. Radiologic studies showed nodules on the septum and/or the turbinates in 14 cases, complete or subtotal opacification of the sinuses and/or the nasal cavities in 13 cases, and nasopharyngeal or pharyngolaryngeal lesions in 4 cases. Treatment with corticosteroids, methotrexate, azathioprine, and surgery appear globally disappointing in view of the side effects and the relapses during a long follow-up (3-15 yr; mean, 6 yr). On the basis of this study, we propose the following diagnostic criteria: 1) histopathologic confirmation of noncaseating granuloma; 2) chronic rhinosinusitis poorly responsive to conventional treatment and radiologic evidence of rhinosinusitis, often with nodules on the septum and/or the turbinates; 3) elevated level of angiotensin-converting enzyme; 4) positive gallium scan (if performed); 5) frequent evidence of systemic, especially pulmonary, sarcoidosis; 6) no evidence of other granulomatous diseases, such as Wegener granulomatosis.
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            The nasal manifestations of sarcoidosis: a review and report of eight cases.

            We report eight patients presenting to the Department of Otorhinolaryngology between 1990 and 1998 in whom a diagnosis of sarcoidosis was made. The most common presenting symptom was that of nasal obstruction and crusting and the most common site of involvement was the septum and inferior turbinate. These patients differ from the majority of patients who present with the other manifestations of sarcoidosis in that they are older. Where mucosal changes are present within the nose, biopsy gives a high diagnostic yield. The aim of treatment is to gain symptomatic control with the lowest dose of steroids. The majority of patient's nasal symptoms were managed with local measures and topical steroids. Nasal disease tends to follow a prolonged but benign course. Few had other organ involvement. While nasal sarcoidosis remains a rare cause of nasal obstruction, biopsy of abnormal nasal mucosa along with further investigations as dictated by history, examination and histological findings is important if delay in diagnosis is to be avoided.
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              Sarcoidosis of the nose and paranasal sinuses.

              Sarcoidosis is a chronic systemic disease of unknown etiology characterized by non-caseating granulomatous inflammation of various organs. The records of 2319 patients with the diagnosis of sarcoidosis were reviewed to determine the incidence of nasal involvement. Seventeen patients or approximately 1% of the patients with sarcoidosis had histologically proven nasal mucosa involvement. These patients had symptoms of nasal crusting, congestion, epistaxis, pain, or anosmia. The clinical findings in these patients included friable nasal mucosa, nasal polyps, or a characteristic submucosal nodularity. Most patients also had abnormal sinus roentgenograms with either thickening of the sinus mucosa or opacification of the sinuses. Biopsy of the nasal mucosa shows typical non-caseating granulomas, but care must be exercised to exclude other causes of granulomatous inflammation of the nasal mucosa including tuberculosis, fungal infections, and other idiopathic granulomatous diseases such as Wegener's granulomatosis and Churg-Strauss syndrome. The treatment of nasal sarcoidosis has consisted of systemic steroids and in some cases topical beclomethasone dipropionate.
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                Author and article information

                Journal
                Diagn Pathol
                Diagnostic Pathology
                BioMed Central (London )
                1746-1596
                2006
                21 November 2006
                : 1
                : 44
                Affiliations
                [1 ]Department of Otorhinolaryngology, Baskent University Faculty of Medicine, Ankara, Turkey
                [2 ]Department of Pathology, Baskent University Faculty of Medicine, Ankara, Turkey
                Article
                1746-1596-1-44
                10.1186/1746-1596-1-44
                1661595
                17118178
                cd33d404-f6f5-434c-a1a0-8e9053533b1f
                Copyright © 2006 Erbek et al; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 3 November 2006
                : 21 November 2006
                Categories
                Case Report

                Pathology
                Pathology

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