Hypereosinophilic syndrome—a rare adverse event of anti-cytokine treatment in rheumatoid arthritis resolved after Janus kinase inhibitor therapy

Šteňová, Emőke; Tarabčáková, Lenka; Babál, Pavel; Kašperová, Stela

doi:10.1007/s10067-020-05134-z

Cited by 10 publications

(8 citation statements)

References 15 publications

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“…7 Furthermore, several other case reports have previously shown encouraging results with tofacitinib for the treatment of eosinophilic fasciitis, eosinophilic esophagitis, and hypereosinophilic syndrome with cutaneous involvement. [8][9][10] Mechanistically, JAK is a rational target in MRH, as proinflammatory IL-6 signals through JAK-1 and elevated IL-6 levels have been described in serum and tissue samples from MRH patients. 11 Moreover, a potentially beneficial role in reducing epithelial inflammation and controlling Th2 responses has been observed with JAK inhibitors.…”

Section: Discussionmentioning

confidence: 99%

Multicentric reticulohistiocytosis with eosinophilic gastroenteritis: An unusual relationship

et al. 2023

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Multicentric reticulohistiocytosis (MRH) is a rare disease with poorly defined therapeutic strategies. Here, we report the case of a patient with eosinophilic gastroenteritis who developed MRH after 5 years. The patient presented with disabling and rapidly progressive polyarthralgia, eosinophilic gastroenteritis, and resistance to first‐line therapies. However, there was a marked improvement in the arthritis symptoms following treatment with tofacitinib. This presentation provides a useful perspective for the therapeutic management of complex scenarios involving MRH.

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Section: Discussionmentioning

confidence: 99%

Multicentric reticulohistiocytosis with eosinophilic gastroenteritis: An unusual relationship

et al. 2023

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“…The findings of this case series suggest that EC may be a T H 2-related inflammatory disease with marked JAK2/pSTAT5 activation, leading to the dysregulated recruitment of eosinophils in the skin. Together with the clinical response to baricitinib, our data advocate for the development of JAK1/JAK2 targeting treatment approaches in EC and other eosinophilic diseases …”

Section: Discussionmentioning

confidence: 99%

“…Together with the clinical response to baricitinib, our data advocate for the development of JAK1/ JAK2 targeting treatment approaches in EC and other eosinophilic diseases. 38,40,41 Data Sharing Statement: See Supplement 2.…”

Section: Discussionmentioning

confidence: 99%

Hyperactivation of the JAK2/STAT5 Signaling Pathway and Evaluation of Baricitinib Treatment Among Patients With Eosinophilic Cellulitis

et al. 2023

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ImportanceThe pathogenesis of eosinophilic cellulitis (EC) is poorly understood, limiting available treatment options. The current treatment paradigm focuses on delayed type 2 hypersensitivity reaction to various triggers.ObjectiveTo gain further insight into the nature of EC inflammation and into the cellular signal transduction pathways that are activated in the context of EC.Design, Setting, and ParticipantsThis case series was conducted in Lyon, France, from January 2018 to December 2021. Analysis of archival skin biopsy samples from patients with EC and from healthy control participants was performed using histology, Janus kinase (JAK)–signal transducer and activator of transcription (STAT) immunohistochemistry, and gene profiling. Data analysis was conducted between January 2020 and January 2022.Main Outcomes and MeasuresPruritus (visual analog score), percentage of body surface area with lesional skin, and RNA transcripts of inflammatory biomarkers from the skin (threshold cycle) were assessed in 1 index patient with refractory EC who received oral JAK1/JAK2 inhibitor baricitinib (4 mg/d).ResultsThis study included samples from 14 patients with EC (7 men and 7 women) and 8 healthy control participants (4 men and 4 women). The mean (SD) age of patients was 52 (20) years. Marked type 2 inflammation (chemokines CCL17, CCL18, and CCL26 and interleukin 13) with preferential activation of the JAK1/JAK2–STAT5 pathways in EC lesions was observed. In the 1 index patient with refractory EC, complete clinical remission of skin lesions was observed after 1 month of treatment with baricitinib.Conclusions and RelevanceThese findings suggest that EC is a type 2 inflammatory disease with preferential activation of the JAK1/JAK2-STAT5 pathways. In addition, these results suggest the potential of treatment approaches targeting JAK1/JAK2 for patients with EC.

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“…A recent study reported that hypereosinophilic syndrome, a rare adverse event of anti-cytokine treatment in RA, was resolved after JAK inhibitor. [ 11 ] Similarly, GM-CSF was reportedly involved in the pathogenesis of RA, and [ 8 ] serum eosinophils by GM-CSF have also been associated with phosphorylation of JAK 2. [ 12 ] These results suggest that induced GM-CSF expression was a common pathological factor between RA and eosinophilic diseases.…”

Section: Discussionmentioning

confidence: 99%

BNT162b2 coronavirus disease-2019 vaccination accelerated rheumatoid arthritis disease activity in chronic eosinophilic pneumonia: A case report

et al. 2022

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Rationale: The relationship between rheumatoid arthritis (RA) and eosinophilic inflammation is unclear. According to recent studies, it has been suggested that T helper 2 cell responses play a role in the inhibition of RA. It is unclear how the immunological response after coronavirus disease-2019 (COVID-19) vaccination affects T cell immune reactions. Patient concerns and diagnoses: Here, we report the case of an 88-year-old woman diagnosed with RA and chronic eosinophilic pneumonia (CEP). She was diagnosed with CEP about 20 years ago, and, through steroid treatment, she improved and had no relapse for 16 years. At the time of diagnosis of CEP, the rheumatoid factor (RF) was increased; however, there were no joint symptoms. After receiving the COVID-19 vaccine, joint and respiratory symptoms gradually worsened. Laboratory examinations showed increased RF, anti-cyclin citrullinated peptide antibody, and peripheral absolute eosinophil count. Musculoskeletal ultrasonography showed synovitis. Intervention and outcome: Methylprednisolone pulse therapy improved respiratory and joint symptoms immediately; RA and CEP stabilized with no relapses. Lessons: Eosinophilic and rheumatoid reactions following COVID-19 vaccination were an-reported adverse events. Eosinophilic inflammation might be reflected on an anti-inflammatory reaction in initial phase of RA.

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Hypereosinophilic syndrome—a rare adverse event of anti-cytokine treatment in rheumatoid arthritis resolved after Janus kinase inhibitor therapy

Cited by 10 publications

References 15 publications

Multicentric reticulohistiocytosis with eosinophilic gastroenteritis: An unusual relationship

Multicentric reticulohistiocytosis with eosinophilic gastroenteritis: An unusual relationship

Hyperactivation of the JAK2/STAT5 Signaling Pathway and Evaluation of Baricitinib Treatment Among Patients With Eosinophilic Cellulitis

BNT162b2 coronavirus disease-2019 vaccination accelerated rheumatoid arthritis disease activity in chronic eosinophilic pneumonia: A case report

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