Empirical use of anakinra in AA amyloidosis of uncertain aetiology

Lane, Thirusha; Rowczenio, DM; Gilbertson, JA; Gillmore, JD; Wechalekar, AD; Hawkins, PN; Lachmann, HJ

doi:10.1186/1546-0096-13-s1-o70

Cited by 2 publications

(4 citation statements)

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“…Tocilizumab was applied intravenously every 2–4 weeks at doses of 8 mg/kg body weight. According to Lane et al, a significant reduction in SAA could be achieved already within 10 days after the first application (70 mg/L initially, 4 mg/L after 10 days) [ 17 ]. Under continued tocilizumab treatment, a regression up to the complete disappearance of the gastrointestinal amyloid deposits has been observed earlier [ 17 , 18 ].…”

Section: Discussionmentioning

confidence: 99%

“…According to Lane et al, a significant reduction in SAA could be achieved already within 10 days after the first application (70 mg/L initially, 4 mg/L after 10 days) [ 17 ]. Under continued tocilizumab treatment, a regression up to the complete disappearance of the gastrointestinal amyloid deposits has been observed earlier [ 17 , 18 ]. Similarly, favorable renal response as indicated by a significant reduction in proteinuria up to complete normalization has been reported [ 18 , 19 ], as well as a reduction in serum creatinine, especially in severe cases (patient 8 in the case study of Yilmaz et al [ 19 ]).…”

Section: Discussionmentioning

confidence: 99%

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Vector autoregression: Useful in rare diseases?—Predicting organ response patterns in a rare case of secondary AA amyloidosis

et al. 2023

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Background Statistical analyses of clinical data are a cornerstone in understanding pathomechanisms of disorders. In rare disorders, cross-sectional datasets of sufficient size are usually not available. Taking AA amyloidosis as an example of a life-threatening rare disorder resulting from of uncontrolled chronic inflammation, we propose techniques from time series analysis to predict organ response to treatment. The advantage of time-series analysis is that it solely relies on temporal variation and therefore allows analyzing organ response to treatment even when the cross-sectional dimension is small. Methods The joint temporal interdependence of inflammatory activity and organ response was modelled multivariately using vector autoregression (VAR) based on a unique 4.5 year spanning data set of routine laboratory, imaging data (e.g., 18F-Florbetaben-PET/CT) and functional investigations of a 68-year-old patient with multi-organ involvement of AA amyloidosis due to ongoing inflammatory activity of a malignant paraganglioma in stable disease for >20 years and excellent response to tocilizumab). Results VAR analysis showed that alterations in inflammatory activity forecasted alkaline phosphatase (AP). AP levels, but not inflammatory activity at the previous measurement time point predicted proteinuria. Conclusion We demonstrate the feasibility and value of time series analysis for obtaining clinically reliable information when the rarity of a disease prevents conventional prognostic modelling approaches. We illustrate the comparative utility of blood, functional and imaging markers to monitor the development and regression of AA amyloidosis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Vector autoregression: Useful in rare diseases?—Predicting organ response patterns in a rare case of secondary AA amyloidosis

et al. 2023

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“…Many patients achieved remission or improvement of AA amyloidosis after the administration of several different biologics. For example, seven patients, who were treated with etanercept, adalimumab, or rituximab, improved and had SAA suppression after tocilizumab therapy, and one patient, who was treated with a TNF inhibitor and tocilizumab, achieved remission with abatacept [30,42] . This suggests that biologic agents need to be replaced to suppress the progression of AA amyloidosis secondary to RA, depending on the patient.…”

Section: Discussionmentioning

confidence: 99%

“…A 77-year-old female with massive ascites and bloody stool was diagnosed with AA amyloidosis after AA depositions were detected in her rectal mucosa, and her ascites disappeared after 5 months of tocilizumab therapy [20] . Tocilizumab is also effective in patients with renal AA amyloidosis, which manifests as proteinuria, hematuria, and renal failure [19,24–31] . Miyagawa et al reported four patients with RA and secondary AA amyloidosis who were treated with tocilizumab, and three had GI tract involvement with proteinuria [28] .…”

Section: Methodsmentioning

confidence: 99%

Biologic therapy for amyloid A amyloidosis secondary to rheumatoid arthritis treated with interleukin 6 therapy

Jung

Kim

et al. 2021

Medicine

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Introduction: Secondary amyloidosis is a rare complication of rheumatoid arthritis (RA) that is histologically characterized by the deposition of amyloid fibrils in target organs, such as the kidneys and gastrointestinal tract. Controlling the inflammatory response is essential to prevent organ dysfunction in amyloid A (AA) amyloidosis secondary to RA, and no clear treatment strategy exists. Patient Concerns and Diagnosis: A 66-year-old woman with RA, who had been treated with disease-modifying anti-rheumatic drugs for 1 year, presented with recurrent abdominal pain and prolonged diarrhea. Endoscopy showed chronic inflammation, and colon tissue histology confirmed AA amyloidosis. Interventions and Outcomes: After tocilizumab therapy was begun, her diarrhea and abdominal pain subsided, and articular symptoms improved. Biologic drugs for RA have been used in patients with secondary AA amyloidosis, including tumor necrosis factor and Janus kinase inhibitors, interleukin 6 blockers, and a T cell modulator. Here, we systematically review existing case reports and compare the outcomes of RA-related AA amyloidosis after treatment with various drugs. Conclusion: The data indicate that biologic drugs like tocilizumab might be treatments of choice for AA amyloidosis secondary to RA.

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Empirical use of anakinra in AA amyloidosis of uncertain aetiology

Cited by 2 publications

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Vector autoregression: Useful in rare diseases?—Predicting organ response patterns in a rare case of secondary AA amyloidosis

Vector autoregression: Useful in rare diseases?—Predicting organ response patterns in a rare case of secondary AA amyloidosis

Biologic therapy for amyloid A amyloidosis secondary to rheumatoid arthritis treated with interleukin 6 therapy

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