Calciphylaxis. A review of 9 cases

Pascual, Leire Loidi; Peñalba, Ana; Jaime, Saioa Oscoz; Iturriagagoitia, Alicia Córdoba; Fraile, Raquel Rodil; Bayona, Juan Ignacio Yanguas

doi:10.1016/j.medcle.2016.09.031

Cited by 4 publications

(3 citation statements)

References 8 publications

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“…Among the 5601 publications retrieved from the targeted databases, 19 retrospective cohort studies 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 (422 patients; mean age, 57 years; 37.3% male) met our eligibility criteria. Among the 422 patients with CKD experiencing calciphylaxis, 347 were dialysis-dependent.…”

Section: Resultsmentioning

confidence: 99%

Intravenous Sodium Thiosulphate for Calciphylaxis of Chronic Kidney Disease

et al. 2023

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ImportanceCalciphylaxis is a rare disease with high mortality mainly involving patients with chronic kidney disease (CKD). Sodium thiosulphate (STS) has been used as an off-label therapeutic in calciphylaxis, but there is a lack of clinical trials and studies that demonstrate its effect compared with those without STS treatment.ObjectiveTo perform a meta-analysis of the cohort studies that provided data comparing outcomes among patients with calciphylaxis treated with and without intravenous STS.Data SourcesPubMed, Embase, Cochrane Library, Web of Science, and ClinicalTrials.gov were searched using relevant terms and synonyms including sodium thiosulphate and calci* without language restriction.Study SelectionThe initial search was for cohort studies published before August 31, 2021, that included adult patients diagnosed with CKD experiencing calciphylaxis and could provide a comparison between patients treated with and without intravenous STS. Studies were excluded if they reported outcomes only from nonintravenous administration of STS or if the outcomes for CKD patients were not provided.Data Extraction and SynthesisRandom-effects models were performed. The Egger test was used to measure publication bias. Heterogeneity was assessed using the I2 test.Main Outcomes and MeasuresSkin lesion improvement and survival, synthesized as ratio data by a random-effects empirical Bayes model.ResultsAmong the 5601 publications retrieved from the targeted databases, 19 retrospective cohort studies including 422 patients (mean age, 57 years; 37.3% male) met the eligibility criteria. No difference was observed in skin lesion improvement (12 studies with 110 patients; risk ratio, 1.23; 95% CI, 0.85-1.78) between the STS and the comparator groups. No difference was noted for the risk of death (15 studies with 158 patients; risk ratio, 0.88; 95% CI, 0.70-1.10) and overall survival using time-to-event data (3 studies with 269 participants; hazard ratio, 0.82; 95% CI, 0.57-1.18). In meta-regression, lesion improvement associated with STS negatively correlated with publication year, implying that recent studies are more likely to report a null association compared with past studies (coefficient = −0.14; P = .008).Conclusions and RelevanceIntravenous STS was not associated with skin lesion improvement or survival benefit in patients with CKD experiencing calciphylaxis. Future investigations are warranted to examine the efficacy and safety of therapies for patients with calciphylaxis.

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Section: Resultsmentioning

confidence: 99%

Intravenous Sodium Thiosulphate for Calciphylaxis of Chronic Kidney Disease

et al. 2023

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“…It includes warfarin-induced skin necrosis, although this usually occurs in the setting of protein C deficiency and usually develops within the first few days of starting warfarin [ 14 ]. Alternatively, calciphylaxis can present with necrotic ulcers although this is usually in the setting of end-stage kidney disease and is usually not seen with moderate chronic kidney disease, as in the present case [ 15 ]. Calciphylaxis can be diagnosed from calcium deposits on biopsy [ 16 ].…”

Section: Discussionmentioning

confidence: 99%

Extensive abdominal wall ulceration as a late manifestation of antiphospholipid syndrome: a case report

2018

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BackgroundAntiphospholipid syndrome is an autoimmune disorder characterized by the presence of antiphospholipid antibodies and commonly presents with vascular thromboembolic phenomena, thrombocytopenia, and obstetric complications. Antiphospholipid syndrome can be classified as either primary or secondary to other connective tissue diseases. Dermatologic manifestations are common; however, non-vasculitic skin ulceration is an uncommon manifestation of antiphospholipid syndrome with limited treatment options.Case presentationIn this paper we report the case of a 58-year-old white woman who developed necrotic abdominal wall ulcers 27 years after a diagnosis of secondary antiphospholipid syndrome associated with systemic lupus erythematosus. The ulcers developed despite our patient being on therapeutic anticoagulation with warfarin and were resistant to further increases in the intensity of anticoagulation. Management was further complicated due to reluctance on the part of our patient to switch over to injectable heparin.ConclusionsThis case highlights a rare late dermatologic presentation of antiphospholipid syndrome, which responded poorly to conventional anticoagulation with warfarin. Current management is limited to experimental therapies and the role of newer anticoagulants is still unknown.

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“…The delay before starting STS treatment also greatly varied within our cohort, which was probably due to limited STS availability at the pharmacy department when clinicians initially started this therapy. Although not statistically significant (probably due to the small number of patients in our cohort), an early STS treatment initiation in calciphylaxis has been advocated as an important contributing factor for treatment success [ 3 , 60 ]. In terms of side effects, 50% of STS treated patients experienced nausea or vomiting but symptoms were not severe enough to justify STS cessation.…”

Section: Key Clinical Features: Therapies Risks Factors and Mortalitymentioning

confidence: 92%

Multimodality approach to treat calciphylaxis in end-stage kidney disease patients

Lajoie,

Ghanemi,

Bourbeau

et al. 2023

Renal Failure

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A multimodality approach has been proposed as an effective treatment for calciphylaxis in patients with end-stage kidney disease. In this retrospective study, we report the cases of 12 end-stage kidney disease patients from l’Hôtel-Dieu de Québec hospital (Canada) who were diagnosed with calciphylaxis between 2004 and 2012 and treated with a multimodality clinical approach including sodium thiosulfate (STS). Statistical analyses were performed to evaluate the impacts of patients characteristics, the different interventions as well as therapy regimen on the therapeutic response. The majority of patients ( n = 9) were hemodialyzed. The patients-associated comorbidities were consistent with previously reported risk factors for calciphylaxis: Diabetes ( n = 11), calcium-based phosphate binders use ( n = 10), warfarin use ( n = 9), obesity ( n = 7), female gender ( n = 8) and intravenous iron use ( n = 8). STS was given for a median duration of 81 days. 75% of the patients had a response (total or partial) including a complete response in 42% of patients. One-year mortality rate was low (25%). STS was used during a mean duration of 83.33 ± 41.52 days and with a total cumulating dose of 1129.00 ± 490.58 g. The recorded mean time before a complete response was 102.20 days (51–143). Pain improvement occurred after a mean time of 8.67 ± 10.06 days. None of the studied factors was statistically associated with a complete or a partial response to the multimodality approach. Although our data have a limited statistical power, they support treating calciphylaxis with a multimodality approach including STS as its effects are independent from important clinical variables.

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Calciphylaxis. A review of 9 cases

Cited by 4 publications

References 8 publications

Intravenous Sodium Thiosulphate for Calciphylaxis of Chronic Kidney Disease

Intravenous Sodium Thiosulphate for Calciphylaxis of Chronic Kidney Disease

Extensive abdominal wall ulceration as a late manifestation of antiphospholipid syndrome: a case report

Multimodality approach to treat calciphylaxis in end-stage kidney disease patients

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