Diagnostic delay of sarcoidosis: an integrated systematic review
Abstract Background Sarcoidosis is a chronic inflammatory granulomatous disease of unknown cause. Delays in diagnosis can result in disease progression and poorer outcomes for patients. Our aim was to review the current literature to determine the overall diagnostic delay of sarcoidosis, factors ass...
| Main Authors: | , , , , , , |
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| Format: | Article |
| Language: | English |
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BMC
2024-04-01
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| Series: | Orphanet Journal of Rare Diseases |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s13023-024-03152-7 |
| _version_ | 1797209043560824832 |
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| author | Tergel Namsrai Christine Phillips Anne Parkinson Dianne Gregory Elaine Kelly Matthew Cook Jane Desborough |
| author_facet | Tergel Namsrai Christine Phillips Anne Parkinson Dianne Gregory Elaine Kelly Matthew Cook Jane Desborough |
| author_sort | Tergel Namsrai |
| collection | DOAJ |
| description | Abstract Background Sarcoidosis is a chronic inflammatory granulomatous disease of unknown cause. Delays in diagnosis can result in disease progression and poorer outcomes for patients. Our aim was to review the current literature to determine the overall diagnostic delay of sarcoidosis, factors associated with diagnostic delay, and the experiences of people with sarcoidosis of diagnostic delay. Methods Three databases (PubMed/Medline, Scopus, and ProQuest) and grey literature sources were searched. Random effects inverse variance meta-analysis was used to pool mean diagnostic delay in all types of sarcoidosis subgroup analysis. Diagnostic delay was defined as the time from reported onset of symptoms to diagnosis of sarcoidosis. Results We identified 374 titles, of which 29 studies were included in the review, with an overall sample of 1531 (694 females, 837 males). The overall mean diagnostic delay in all types of sarcoidosis was 7.93 months (95% CI 1.21 to 14.64 months). Meta-aggregation of factors related to diagnostic delay in the included studies identified three categories: (1) the complex and rare features of sarcoidosis, (2) healthcare factors and (3) patient-centred factors. Meta-aggregation of outcomes reported in case studies revealed that the three most frequent outcomes associated with diagnostic delay were: (1) incorrect diagnosis, (2) incorrect treatment and (3) development of complications/disease progression. There was no significant difference in diagnostic delay between countries with gatekeeper health systems (where consumers are referred from a primary care clinician to specialist care) and countries with non-gatekeeper systems. No qualitative studies examining people’s experiences of diagnostic delay were identified. Conclusion The mean diagnostic delay for sarcoidosis is almost 8 months, which has objective consequences for patient management. On the other hand, there is a paucity of evidence about the experience of diagnostic delay in sarcoidosis and factors related to this. Gaining an understanding of people’s experiences while seeking a diagnosis of sarcoidosis is vital to gain insight into factors that may contribute to delays, and subsequently inform strategies, tools and training activities aimed at increasing clinician and public awareness about this rare condition. Trial registration PROSPERO Registration number: CRD42022307236. |
| first_indexed | 2024-04-24T09:48:26Z |
| format | Article |
| id | doaj.art-24aa4eefbb884d0687d3042d42b87413 |
| institution | Directory Open Access Journal |
| issn | 1750-1172 |
| language | English |
| last_indexed | 2024-04-24T09:48:26Z |
| publishDate | 2024-04-01 |
| publisher | BMC |
| record_format | Article |
| series | Orphanet Journal of Rare Diseases |
| spelling | doaj.art-24aa4eefbb884d0687d3042d42b874132024-04-14T11:29:14ZengBMCOrphanet Journal of Rare Diseases1750-11722024-04-0119111310.1186/s13023-024-03152-7Diagnostic delay of sarcoidosis: an integrated systematic reviewTergel Namsrai0Christine Phillips1Anne Parkinson2Dianne Gregory3Elaine Kelly4Matthew Cook5Jane Desborough6National Centre for Epidemiology and Population Health, The Australian National UniversitySchool of Medicine and Psychology, The Australian National UniversityNational Centre for Epidemiology and Population Health, The Australian National UniversityNational Centre for Epidemiology and Population Health, The Australian National UniversityNational Centre for Epidemiology and Population Health, The Australian National UniversityJohn Curtin School of Medical Research, The Australian National UniversityNational Centre for Epidemiology and Population Health, The Australian National UniversityAbstract Background Sarcoidosis is a chronic inflammatory granulomatous disease of unknown cause. Delays in diagnosis can result in disease progression and poorer outcomes for patients. Our aim was to review the current literature to determine the overall diagnostic delay of sarcoidosis, factors associated with diagnostic delay, and the experiences of people with sarcoidosis of diagnostic delay. Methods Three databases (PubMed/Medline, Scopus, and ProQuest) and grey literature sources were searched. Random effects inverse variance meta-analysis was used to pool mean diagnostic delay in all types of sarcoidosis subgroup analysis. Diagnostic delay was defined as the time from reported onset of symptoms to diagnosis of sarcoidosis. Results We identified 374 titles, of which 29 studies were included in the review, with an overall sample of 1531 (694 females, 837 males). The overall mean diagnostic delay in all types of sarcoidosis was 7.93 months (95% CI 1.21 to 14.64 months). Meta-aggregation of factors related to diagnostic delay in the included studies identified three categories: (1) the complex and rare features of sarcoidosis, (2) healthcare factors and (3) patient-centred factors. Meta-aggregation of outcomes reported in case studies revealed that the three most frequent outcomes associated with diagnostic delay were: (1) incorrect diagnosis, (2) incorrect treatment and (3) development of complications/disease progression. There was no significant difference in diagnostic delay between countries with gatekeeper health systems (where consumers are referred from a primary care clinician to specialist care) and countries with non-gatekeeper systems. No qualitative studies examining people’s experiences of diagnostic delay were identified. Conclusion The mean diagnostic delay for sarcoidosis is almost 8 months, which has objective consequences for patient management. On the other hand, there is a paucity of evidence about the experience of diagnostic delay in sarcoidosis and factors related to this. Gaining an understanding of people’s experiences while seeking a diagnosis of sarcoidosis is vital to gain insight into factors that may contribute to delays, and subsequently inform strategies, tools and training activities aimed at increasing clinician and public awareness about this rare condition. Trial registration PROSPERO Registration number: CRD42022307236.https://doi.org/10.1186/s13023-024-03152-7SarcoidosisDiagnostic delayMisdiagnosisSystematic reviewMeta-analysisMeta-aggregation |
| spellingShingle | Tergel Namsrai Christine Phillips Anne Parkinson Dianne Gregory Elaine Kelly Matthew Cook Jane Desborough Diagnostic delay of sarcoidosis: an integrated systematic review Orphanet Journal of Rare Diseases Sarcoidosis Diagnostic delay Misdiagnosis Systematic review Meta-analysis Meta-aggregation |
| title | Diagnostic delay of sarcoidosis: an integrated systematic review |
| title_full | Diagnostic delay of sarcoidosis: an integrated systematic review |
| title_fullStr | Diagnostic delay of sarcoidosis: an integrated systematic review |
| title_full_unstemmed | Diagnostic delay of sarcoidosis: an integrated systematic review |
| title_short | Diagnostic delay of sarcoidosis: an integrated systematic review |
| title_sort | diagnostic delay of sarcoidosis an integrated systematic review |
| topic | Sarcoidosis Diagnostic delay Misdiagnosis Systematic review Meta-analysis Meta-aggregation |
| url | https://doi.org/10.1186/s13023-024-03152-7 |
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