Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study.
<h4>Background</h4>Sinonasal respiratory epithelial adenomatoid hamartomas (REAH) are rare benign lesions with diverse clinical characteristics. Their pathogenesis remains unclear, with hypotheses suggesting either an inflammatory process or neoplastic origin. Despite their clinical hete...
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2025-01-01
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| Online Access: | https://doi.org/10.1371/journal.pone.0318452 |
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| author | Yujin Heo Sang Duk Hong Hyo Yeol Kim Gwanghui Ryu Kyung Won Kwon Yong Gi Jung |
| author_facet | Yujin Heo Sang Duk Hong Hyo Yeol Kim Gwanghui Ryu Kyung Won Kwon Yong Gi Jung |
| author_sort | Yujin Heo |
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| description | <h4>Background</h4>Sinonasal respiratory epithelial adenomatoid hamartomas (REAH) are rare benign lesions with diverse clinical characteristics. Their pathogenesis remains unclear, with hypotheses suggesting either an inflammatory process or neoplastic origin. Despite their clinical heterogeneity, a formal subtype classification is lacking. This study analyzes the clinical features of surgically treated sinonasal REAH and investigates disease characteristics based on bilaterality, particularly in relation to type 2 chronic rhinosinusitis.<h4>Methods</h4>This retrospective analysis included patients with REAH who underwent endoscopic surgery and received pathological confirmation between November 2008 and November 2023. Demographic data, including asthma history, prior sinus surgeries, serum eosinophil levels, and the Japanese Epidemiological Survey of Refractory Eosinophilic Chronic Rhinosinusitis (JESREC) score, were analyzed in relation to REAH bilaterality.<h4>Results</h4>Among 21 patients (17 males, 4 females; mean age 46.5 years), 11 cases (52.4%) exhibited bilateral origins (REAH_bi) in the superior nasal septum and olfactory cleft, while 10 cases (47.6%) presented as isolated unilateral lesions (REAH_uni). REAH_bi was significantly associated with asthma (p = 0.012), prior sinus surgery (p = 0.002), and inflammatory polyposis (p = 0.002) compared to REAH_uni. Elevated preoperative serum eosinophil levels and JESREC scores were also noted in REAH_bi cases (p = 0.021 and <0.001, respectively). Neither group showed recurrence during a mean follow-up of 11.91 months.<h4>Conclusion</h4>Bilaterally originating REAH in the superior nasal septum and olfactory cleft demonstrates pronounced type 2 inflammatory characteristics, suggesting potential differences in pathogenesis compared to unilateral REAH. These findings underscore the need for further investigation into REAH pathophysiology and emphasize the importance of bilaterality in clinical assessment. |
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| institution | Kabale University |
| issn | 1932-6203 |
| language | English |
| publishDate | 2025-01-01 |
| publisher | Public Library of Science (PLoS) |
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| spelling | doaj-art-114df1fff8f442a8aa27d4e8a7f732332025-02-07T05:30:56ZengPublic Library of Science (PLoS)PLoS ONE1932-62032025-01-01202e031845210.1371/journal.pone.0318452Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study.Yujin HeoSang Duk HongHyo Yeol KimGwanghui RyuKyung Won KwonYong Gi Jung<h4>Background</h4>Sinonasal respiratory epithelial adenomatoid hamartomas (REAH) are rare benign lesions with diverse clinical characteristics. Their pathogenesis remains unclear, with hypotheses suggesting either an inflammatory process or neoplastic origin. Despite their clinical heterogeneity, a formal subtype classification is lacking. This study analyzes the clinical features of surgically treated sinonasal REAH and investigates disease characteristics based on bilaterality, particularly in relation to type 2 chronic rhinosinusitis.<h4>Methods</h4>This retrospective analysis included patients with REAH who underwent endoscopic surgery and received pathological confirmation between November 2008 and November 2023. Demographic data, including asthma history, prior sinus surgeries, serum eosinophil levels, and the Japanese Epidemiological Survey of Refractory Eosinophilic Chronic Rhinosinusitis (JESREC) score, were analyzed in relation to REAH bilaterality.<h4>Results</h4>Among 21 patients (17 males, 4 females; mean age 46.5 years), 11 cases (52.4%) exhibited bilateral origins (REAH_bi) in the superior nasal septum and olfactory cleft, while 10 cases (47.6%) presented as isolated unilateral lesions (REAH_uni). REAH_bi was significantly associated with asthma (p = 0.012), prior sinus surgery (p = 0.002), and inflammatory polyposis (p = 0.002) compared to REAH_uni. Elevated preoperative serum eosinophil levels and JESREC scores were also noted in REAH_bi cases (p = 0.021 and <0.001, respectively). Neither group showed recurrence during a mean follow-up of 11.91 months.<h4>Conclusion</h4>Bilaterally originating REAH in the superior nasal septum and olfactory cleft demonstrates pronounced type 2 inflammatory characteristics, suggesting potential differences in pathogenesis compared to unilateral REAH. These findings underscore the need for further investigation into REAH pathophysiology and emphasize the importance of bilaterality in clinical assessment.https://doi.org/10.1371/journal.pone.0318452 |
| spellingShingle | Yujin Heo Sang Duk Hong Hyo Yeol Kim Gwanghui Ryu Kyung Won Kwon Yong Gi Jung Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. PLoS ONE |
| title | Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. |
| title_full | Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. |
| title_fullStr | Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. |
| title_full_unstemmed | Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. |
| title_short | Link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas: A two-center retrospective cohort study. |
| title_sort | link between bilaterality and type 2 inflammation in respiratory epithelial adenomatoid hamartomas a two center retrospective cohort study |
| url | https://doi.org/10.1371/journal.pone.0318452 |
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