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      Development of CT-based methods for longitudinal analyses of paranasal sinus osteitis in granulomatosis with polyangiitis

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          Abstract

          Background

          Even though progressive rhinosinusitis with osteitis is a major clinical problem in granulomatosis with polyangiitis (GPA), there are no studies on how GPA-related osteitis develops over time, and no quantitative methods for longitudinal assessment.

          Here, we aimed to identify simple and robust CT-based methods for capture and quantification of time-dependent changes in GPA-related paranasal sinus osteitis and compare performance of the methods under study in a largely unselected GPA cohort.

          Methods

          GPA patients (n = 121) with ≥3 paranasal CT scans obtained ≥12 months apart and control patients not having GPA or rhinosinusitis (n = 15) were analysed by: (i) Global osteitis scoring scale (GOSS), originally developed for chronic rhinosinusitis; (ii) Paranasal sinus volume by manual segmentation; (iii) Mean maxillary and sphenoid diameter normalised to landmark distances (i.e. diameter ratio measurement, DRM).

          Results

          Time-dependent changes in GPA-related osteitis were equally well measured by the simple DRM and the labour-intensive volume method while GOSS missed ongoing changes in cases with extensive osteitis. GOSS at last CT combined with DRM identified three distinct patient groups: (i) The no osteitis group, who had no osteitis and no change in DRM from baseline CT to last CT (45/121 GPA patients and 15/15 disease controls); (ii) Stable osteitis group, with presence of osteitis, but no change in DRM across time (31 GPA); (iii) Progressive osteitis, defined by declining DRM (45 GPA).

          Conclusions

          We suggest DRM and GOSS as complementary methods for capturing, classifying and quantifying time-dependent changes in GPA-related osteitis.

          Electronic supplementary material

          The online version of this article (10.1186/s12880-019-0315-7) contains supplementary material, which is available to authorized users.

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          Most cited references22

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          Wegener granulomatosis: an analysis of 158 patients.

          To prospectively study the clinical features, pathophysiology, treatment and prognosis of Wegener granulomatosis. Of the 180 patients with Wegener granulomatosis referred to the National Institute of Allergy and Infectious Diseases during the past 24 years, 158 have been followed for 6 months to 24 years (a total of 1229 patient-years). Characteristics of clinical presentation, surgical pathology, course of illness, laboratory and radiographic findings, and the results of medical and surgical treatment have been recorded in a computer-based information retrieval system. The Warren Magnuson Clinical Center of the National Institutes of Health. Men and women were equally represented; 97% of patients were white, and 85% were more than 19 years of age. The mean period of follow-up was 8 years. One hundred and thirty-three patients (84%) received "standard" therapy with daily low-dose cyclophosphamide and glucocorticoids. Eight (5.0%) received only low-dose cyclophosphamide. Six (4.0%) never received cyclophosphamide and were treated with other cytotoxic agents and glucocorticoids. Ten patients (6.0%) were treated with only glucocorticoids. Ninety-one percent of patients experienced marked improvement, and 75% achieved complete remission. Fifty percent of remissions were associated with one or more relapses. Of 99 patients followed for greater than 5 years, 44% had remissions of greater than 5 years duration. Thirteen percent of patients died of Wegener granulomatosis, treatment-related causes, or both. Almost all patients had serious morbidity from irreversible features of their disease (86%) or side effects of treatment (42%). The course of Wegener granulomatosis has been dramatically improved by daily treatment with cyclophosphamide and glucocorticoids. Nonetheless, disease- and treatment-related morbidity is often profound. Alternative forms of therapy have not yet achieved the high rates of remission induction and successful maintenance that have been reported with daily cyclophosphamide treatment. Despite continued therapeutic success with cyclophosphamide, our long-term follow-up of patients with Wegener granulomatosis has led to increasing concerns about toxicity resulting from prolonged cyclophosphamide therapy and has encouraged investigation of other therapeutic regimens.
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            Staging in rhinosinusitus.

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              Prevalence of Wegener's granulomatosis, microscopic polyangiitis, polyarteritis nodosa and Churg-Strauss syndrome within a defined population in southern Sweden.

              To estimate the point prevalence (p.p.) of Wegener's granulomatosis (WG), microscopic polyangiitis (MPA), polyarteritis nodosa (PAN) and Churg-Strauss syndrome (CSS) within a defined population in southern Sweden. A cross-sectional p.p. study using multiple sources for case identification. The study area, a healthcare district around the city of Lund in southern Sweden (Mellersta Skånes sjukvårdsdistrikt), had, on 31 December 2002, a total population of 287 479 inhabitants. All the identified cases were verified by medical record review. The patients were classified according to an algorithm based on the American College of Rheumatology classification criteria 1990 and the Chapel Hill Consensus Conference definitions 1994. Eighty-six patients (49% female) with a median age of 64.8 yrs (range 15-90.5) fulfilled the study criteria. There were 46 patients with WG; 27 with MPA; nine with PAN; and four with CSS. The p.p. per million inhabitants was estimated on 1 January 2003 to be 160 (95% confidence interval 114-206) for WG, 94 (58-129) for MPA, 31 (11-52) for PAN and 14 (0.3-27) for CSS. Capture-recapture analysis estimated the completeness of the case finding to 96%. The prevalence of WG, MPA, PAN and CSS in our district is the highest figure reported so far. Explanations for this finding may include high incidence, extensive ANCA-testing, good survival as well as sensitive search methods for case identification.
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                Author and article information

                Contributors
                sigrun.skaar.holme@gmail.com
                jonmoe@ous-hf.no
                karin.kilian@medisin.uio.no
                hilde.haukeland@mhh.no
                oyvind.molberg@medisin.uio.no
                h.b.eggesbo@medisin.uio.no
                Journal
                BMC Med Imaging
                BMC Med Imaging
                BMC Medical Imaging
                BioMed Central (London )
                1471-2342
                4 February 2019
                4 February 2019
                2019
                : 19
                : 13
                Affiliations
                [1 ]ISNI 0000 0004 0389 8485, GRID grid.55325.34, Division of Radiology and Nuclear Medicine, Oslo University Hospital, ; PB 4950 Nydalen, Oslo, 0424 Norway
                [2 ]ISNI 0000 0004 1936 8921, GRID grid.5510.1, Institute of Clinical Medicine, University of Oslo, ; PB 1072 Blindern, Oslo, 0316 Norway
                [3 ]Department of Rheumatology, Dermatology and Infectious Diseases, PB 4950 Nydalen, Oslo, 0424 Norway
                [4 ]Department of Rheumatology, Martina Hansen Hospital, Dønskiveien 8, Gjettum, 1346 Norway
                Author information
                http://orcid.org/0000-0001-8713-3639
                Article
                315
                10.1186/s12880-019-0315-7
                6360792
                30717680
                cd5b8af7-c1f7-4c10-87fb-e4a4633da3dd
                © The Author(s) 2019

                Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 24 November 2018
                : 17 January 2019
                Funding
                Funded by: FundRef http://dx.doi.org/10.13039/100009471, EkstraStiftelsen Helse og Rehabilitering;
                Award ID: 2015FO76400
                Categories
                Research Article
                Custom metadata
                © The Author(s) 2019

                Radiology & Imaging
                osteitis,granulomatosis with polyangiitis,paranasal sinuses,ct,longitudinal studies

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