Rosai–Dorfman‐type histiocytes occur in eosinophilic chronic rhinosinusitis and potentially play a role in disease initiation and persistence

Wilsher, Mark James; Bonar, S. Fiona

doi:10.1111/his.12981

Cited by 2 publications

(9 citation statements)

References 23 publications

(72 reference statements)

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“…These findings appear virtually identical to cases previously reported as RDD-like histiocytes. 23 Our findings also validate the clinical significance of this histological pattern. Patients with histological features of RDD demonstrated severe pan-sinusitis symptoms and had a substantially higher need for repeat surgery compared to chronic sinusitis patients without these findings.…”

Section: Discussionsupporting

confidence: 77%

“…Despite the relatively limited volume of histiocytes compared to conventional RDD, they were a prominent feature throughout all cases that starkly differentiated them from other chronic sinusitis specimens. As this impression differed from the previous report of RDD-like histiocytes, which also were seen at higher prevalence, 23 it is possible that we selected more florid cases. Moreover, although no cases had MAP2K1 or KRAS mutations characteristic of conventional RDD, two harboured oncogenic alterations in NF1 and KEAP1.…”

Section: Discussioncontrasting

confidence: 73%

“…A group of nearly identical cases were previously described as RDD-like histiocytes because the overall clinicopathological picture was of chronic sinusitis, rather than RDD. 23 Indeed, several features noted in this series also support separate classification. First, the abnormal histiocytes are quantitatively and qualitatively limited.…”

Section: Discussionsupporting

confidence: 61%

“…These clinical features also parallel cases reported as RDD-like histiocytes. 23 Notably, these findings were seen in 3.5% of all sinus contents specimens over 2 years, suggesting that this is an uncommon but recurrent phenomenon. This histological pattern also appears to be under-recognised, as all cases identified retrospectively were originally signed out as variations on chronic or allergic sinusitis.…”

Section: Discussionmentioning

confidence: 88%

“…[16][17][18][19][20][21][22] Recently, understanding of sinonasal RDD became more complicated with description of RDD-like histiocytesa phenomenon defined by the presence of histiocyte aggregates with histological features of RDD in patients with chronic sinusitis symptoms. 23 It is currently unclear whether these histiocytes truly represent a separate process or a localised manifestation of RDD. In this study, we aimed to further characterise the clinicopathological and molecular features of this phenomenon to clarify its prevalence, classification and clinical implications.…”

Section: Introductionmentioning

confidence: 99%

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Limited sinonasal Rosai–Dorfman disease presenting as chronic sinusitis

et al. 2022

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Aims The sinonasal tract is a common extranodal site for Rosai–Dorfman disease (RDD). Recently, histiocytes with features of RDD were identified in the clinical setting of chronic sinusitis. This study evaluates whether this phenomenon should be considered part of the RDD spectrum or classified separately as RDD‐like histiocytes. Methods and results We prospectively collected 13 cases showing histological features of RDD in chronic sinusitis patients and identified 14 with similar findings (3.5%) via retrospective review of 403 sinus contents over 2 years. All 27 cases displayed nodular aggregates of eosinophilic histiocytes with intermixed lymphoplasmacytic inflammation, prominent eosinophils and emperipolesis. The histiocytes were positive for S100 protein and cyclin D1 and negative for CD1a and CD207. All patients presented with severe chronic sinusitis without tumour formation or systemic symptoms. Twelve patients with follow‐up (55%) required repeat sinus surgery compared with just 43 other sinusitis patients evaluated (11%); features of RDD were present in their additional specimens. Two cases that underwent targeted next‐generation sequencing (20%) had oncogenic mutations in NF1 and KEAP1. Conclusions Overall, these findings confirm diagnostic histological and immunohistochemical features of RDD in a subset of chronic sinusitis specimens. While patients uniformly lack systemic involvement or tumefactive growth, they have a high risk of recurrent sinus disease. Although the relatively subtle nature of the findings raises consideration of separate classification, the presence of occasional oncogenic mutations and evidence of consistent MAPK/ERK pathway activation via cyclin D1 positivity suggests that this phenomenon represents a unique limited manifestation of RDD.

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Section: Discussionsupporting

confidence: 77%

Section: Discussioncontrasting

confidence: 73%

Section: Discussionsupporting

confidence: 61%

Section: Discussionmentioning

confidence: 88%