Non‐uraemic calciphylaxis successfully treated with pamidronate infusion

Truong, David; Riedhammer, Marcus M.; Zinszer, Kathya

doi:10.1111/iwj.13019

Cited by 10 publications

(7 citation statements)

References 25 publications

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“…Table 3 summarises the existing published case reports of NUC treated with the IV medications pamidronate, zoledronate, and sodium thiosulfate (STS), as well as intralesional STS. Two studies using IV pamidronate reported complete closure after 6 months of treatment 26,28 . One study using IV pamidronate did not report the outcome of complete closure 28 .…”

Section: Discussionmentioning

confidence: 99%

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Topical cannabis‐based medicines – A novel paradigm and treatment for non‐uremic calciphylaxis leg ulcers: An open label trial

Maida

Shi

Fazzari

et al. 2020

International Wound Journal

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Non‐Uremic Calciphylaxis (NUC) is a rare condition that often manifests as intractable and painful integumentary wounds, afflicting patients with a high burden of co‐morbidity. The Endocannabinoid System (ECS) is a ubiquitous signalling system that is theorised to be dysregulated within wound beds and associated peri‐wound tissues. Preclinical research has shown that the dominant chemical classes derived from the cannabis plant, cannabinoids, terpenes, and flavonoids, interact with the integumentary ECS to promote wound closure and analgesia. This is a prospective open label cohort study involving two elderly Caucasian females with recalcitrant NUC leg ulcers of greater than 6 months duration. Topical Cannabis‐Based Medicines (TCBM) composed of cannabinoids, terpenes, and flavonoids were applied daily to both the wound bed and peri‐wound tissues until complete wound closure was achieved. Wounds were photographed regularly, and the digital images were subjected to planimetric analysis to objectively quantify the degree of granulation and epithelization. Analgesic utilisation, as a surrogate/proxy for pain scores, was also tracked. The cohort had a mean M3 multimorbidity index score of 3.31. Complete wound closure was achieved in a mean of 76.3 days. Additionally, no analgesics were required after a mean of 63 days. The treatments were well tolerated with no adverse reactions. The positive results demonstrated in very challenging wounds such as NUC, among highly complex patients, suggest that TCBM may have an even broader role within integumentary and wound management. This treatment paradigm warrants being trialled in other wound types and classes, and ultimately should be subjected to randomised controlled trials.

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Section: Discussionmentioning

confidence: 99%

“…Two studies using IV pamidronate reported complete closure after 6 months of treatment. 26,28 One study using IV pamidronate did not report the outcome of complete closure. 28 The report of zoledronate treatment did not report the outcome of complete closure.…”

Section: Discussionmentioning

confidence: 99%

Topical cannabis‐based medicines – A novel paradigm and treatment for non‐uremic calciphylaxis leg ulcers: An open label trial

Maida

Shi

Fazzari

et al. 2020

International Wound Journal

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“…The management of CUA remains problematic as clinicians have largely relied on case series and small uncontrolled studies for guidance. Several agents such as sodium thiosulphate, calcimimetics [ 5 ], bisphosphonates [ 6 ] and vitamin K have been identified as showing promise in small studies, although real benefit of these agents in isolation is still under debate [ 7 – 9 ]. Country-based registries and studies have been a major resource in providing data on the natural history and outcomes of CUA [ 10 ].…”

Section: Introductionmentioning

confidence: 99%

Calciphylaxis in end-stage kidney disease: outcome data from the United Kingdom Calciphylaxis Study

et al. 2021

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Background and aims Calciphylaxis is a rare condition associated with very high mortality in patients with end-stage kidney disease. Data from country-based registries have been an invaluable resource for a better understanding of the natural history and management for this condition. This study aimed to investigate the current management strategies and outcomes of patients enrolled in the United Kingdom Calciphylaxis study (UKCS). Methods The study was conducted on 89 patients registered in the UKCS since 2012. The initial analysis included a description of the baseline characteristics, management strategies and outcomes on follow-up until May 2020. Further analysis included a comparison of the mortality outcome of the UKCS patients who were receiving haemodialysis with a propensity score matched cohort of haemodialysis patients from the Chronic Renal Insufficiency Standards Implementation Study- Haemodialysis (CRISIS-HD). Results Median age of the cohort was 59 years, with a predominance of females (61%) and Caucasian (95%) ethnicity. About 54% of the patients were diabetic and 70% were receiving haemodialysis at study entry. The skin lesions were mostly distributed in the lower extremities (48%). Sodium thiosulphate and calcimimetic were the most widely used management strategies. The mortality rate was 72 deaths per hundred patient-years (50 deaths observed in 69.5 patient years). Complete wound healing was noted in 17% and bacteraemia was reported in 26% of patients. In a comparative analysis of the matched haemodialysis patients, the presence of calciphylaxis in 62 patients showed a strong association with all-cause mortality (HR 6.96; p < 0.001), with annual mortality 67% versus 10.2% in haemodialysis patients without calciphylaxis. Conclusions This UK wide study strengthens the evidence that calciphylaxis is a strong and independent risk factor associated with all-cause mortality; no significant benefit was shown with any individual treatment modality. Until further evidence becomes available, a multifaceted approach would be the appropriate treatment strategy in the management of this extremely serious condition. Graphic abstract

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“…73 The effective use of bisphosphonates has been reported in case series of patients with uremic as well as with non-uremic calciphylaxis. [74][75][76] However, long-term use of bisphosphonates in dialysis patients is usually avoided because of their strong influence on bone metabolism, so any treatment with bisphosphonates should be discussed in an interdisciplinary setting. Another interesting compound for the treatment of calciphylaxis is sodium thiosulfate.…”

Section: Treatmentmentioning

confidence: 99%

Occlusive cutaneous vasculopathies as cause of chronic ulcers

Ronicke,

Berking,

Erfurt‐Berge

2024

J Deutsche Derma Gesell

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SummaryThe term occluding vasculopathies covers a large number of different conditions. These often manifest as skin ulcers. Occluding vasculopathies should be considered in the differential diagnosis of leg ulcers. The term “occlusive vasculopathies” encompasses pathophysiologically related entities that share structural or thrombotic obliteration of small cutaneous vessels. In this article, we will focus on livedoid vasculopathy with and without antiphospholipid syndrome and calciphylaxis with differentiation from hypertonic leg ulcer as the most relevant differential diagnoses of leg ulcer. The term also includes vascular occlusion, for example due to oxalate or cholesterol embolism, and septic vasculopathy. This often leads to acral ulceration and is therefore not a differential diagnosis with classic leg ulcers. It will not be discussed in this article.Occlusive vasculopathy may be suspected in the presence of the typical livedo racemosa or (non‐inflammatory) retiform purpura as a sign of reduced cutaneous perfusion in the wound area. Inflammatory dermatoses, especially vasculitides, must be differentiated. This is achieved by histopathological evaluation of a tissue sample of sufficient size and depth taken at the appropriate time. In addition, specific laboratory parameters, particularly coagulation parameters, can support the diagnosis.

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Non‐uraemic calciphylaxis successfully treated with pamidronate infusion

Cited by 10 publications

References 25 publications

Topical cannabis‐based medicines – A novel paradigm and treatment for non‐uremic calciphylaxis leg ulcers: An open label trial

Topical cannabis‐based medicines – A novel paradigm and treatment for non‐uremic calciphylaxis leg ulcers: An open label trial

Calciphylaxis in end-stage kidney disease: outcome data from the United Kingdom Calciphylaxis Study

Occlusive cutaneous vasculopathies as cause of chronic ulcers

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