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Scientific letter
Non-uremic calciphylaxis: Case series
Calcifilaxis no urémica: descripción de una serie de casos
Carlos César Martí Pelluch
Corresponding author
ccmarti@fphag.org

Corresponding author.
, Anna Soler Sendra, Mònica Vila Vall-llovera
Servicio de Medicina Interna, Hospital General de Granollers, Granollers, Barcelona, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Calciphylaxis is a rare disease&#44; consisting of progressive vascular calcification of small and medium calibre arteries&#44; with endothelial proliferation&#44; fibrosis&#44; thrombosis and skin necrosis with painful ulcers&#46; In advanced chronic kidney disease &#40;ACKD&#41;&#44; the phospho-calcium imbalance present is directly related to the development of lesions&#46; However&#44; in the case of non-uremic calciphylaxis &#40;NUC&#41;&#44; only several risk factors have been identified&#44; choosing to extrapolate the treatment of uremic calciphylaxis &#40;UC&#41;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a>&#46; While UC and NUC are known for high mortality rates &#40;up to 52&#37;&#41;&#44; a number of recent NUC cases have been published with higher survival rates<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#8211;5</span></a>&#46; In this paper we report our experience with 12 cases&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A retrospective study of cases diagnosed with NUC at the General Hospital of Granollers&#44; between April 2013 and December 2021&#44; was conducted&#46; The inclusion criteria were patients with biopsied lesions with a diagnosis of calciphylaxis&#46; Patients with ACKD were excluded&#44; as well as those with loss to follow-up&#46; Demographic data&#44; risk factors described in the literature and progression under treatment with sodium thiosulfate &#40;25&#8239;mg intravenous&#47;3 times per week&#41;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a> were collected&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Fifteen cases were identified in the period described&#46; Three were discarded due to loss to follow-up&#46; Of the 12 patients included&#44; all were Caucasian&#44; 66&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;8&#41; were female&#44; with a median age of 78&#46;4 years &#40;IQR&#58; 71&#46;6-85 years&#41;&#46; 66&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;8&#41; were obese &#40;BMI&#58; 34&#46;2&#8239;kg&#47;m<span class="elsevierStyleSup">2</span> &#91;IQR&#58; 24&#46;2&#8211;40&#46;2&#8239;kg&#47;m<span class="elsevierStyleSup">2</span>&#93;&#41;&#46; The median glomerular filtration rate was 53&#46;5&#8239;ml&#47;min&#47;1&#46;73&#8239;m<span class="elsevierStyleSup">2</span> &#40;IQR&#58; 37&#46;5&#8211;62&#46;5&#8239;ml&#47;min&#47;1&#46;73&#8239;m<span class="elsevierStyleSup">2</span>&#41;&#46; All were multi-pathological&#44; 100&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;12&#41; hypertensive and 66&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;8&#41; were type 2 diabetic&#46; 33&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;4&#41; had a systemic disease &#40;one psoriatic arthritis&#44; 2 rheumatoid arthritis&#44; one lupus&#41;&#44; none of them had liver disease or secondary hyperparathyroidism&#46; 8&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;1&#41; had osteoporosis&#46; None had coagulopathies&#44; and only 8&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;1&#41; were&#44; diagnosed with tumour disease &#40;prostate adenocarcinoma&#41;&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">No abnormalities in calcaemia&#44; phosphatemia or alkaline phosphatase were detected&#46; 50&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;6&#41; had malnutrition parameters &#40;median albumin of 3&#46;4&#8239;g&#47;dl &#40;IQR&#58; 2&#46;8&#8211;3&#46;9&#8239;g&#47;dl&#41;&#46; Regarding concomitant treatments&#59; 50&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;6&#41; were following treatment with acenocoumarol&#44; 41&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;5&#41; chronic corticotherapy&#44; 83&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;12&#41; vitamin D supplementation and 16&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;2&#41; oral iron&#46; All had lesions in the lower limbs&#46; Some 16&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;2&#41; required dose adjustment&#44; with metabolic acidosis being the most common side effect &#40;83&#46;3&#37;&#41;&#46; The anticoagulant drug was changed for all those previously treated with acenocoumarol&#46; Some 58&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;7&#41; had complete resolution &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; 25&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;3&#41; lesion improvement&#44; 8&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;1&#41; experienced stability&#44; and only 8&#46;3&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;1&#41; experienced a clinical deterioration of the lesions&#46; The median treatment duration was 20&#46;7 weeks &#40;IQR&#58; 18-22&#46;7 weeks&#41;&#46; Total mortality was 41&#46;7&#37; &#40;<span class="elsevierStyleItalic">n</span>&#8239;&#61;&#8239;5&#41;&#44; 2 related to NUC &#40;septic shock&#41; and 3 others secondary to decompensation of the underlying disease&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Among our results&#44; we highlight the fact that all patients had more than one concomitant factor described for NUC<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a>&#44; in addition to being multi-pathological&#59; raising suspicion of a multifactorial aetiology&#46; Although Bajaj et al&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> highlight the relationship between the high prevalence of patients with vitamin K antagonists&#44; the fact that 57&#46;1&#37; of our patients were not treated with these drugs casts doubt on whether this is a determining factor in the pathogenesis&#46; All had lower limb ulcers&#44; suggesting a probable anatomical predisposing factor&#44; such as venous return&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Despite the fact that UC involves a phospho-calcium imbalance&#44; all our subjects showed normality in these parameters&#46; This is why we believe that its determination should not constitute a diagnostic mainstay&#46; We highlight our positive experience with sodium thiosulfate&#44; observing that most patients obtained complete resolution&#44; along with lesion healing&#46; It is important to highlight that the cure rate is biased by the high mortality rate since&#44; out of 5 deaths&#44; only 2 of them were directly related to NUC&#44; the rest were linked to the decompensation of underlying diseases&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; NUC is a poorly studied disease with few articles clarifying its pathophysiology&#46; Treatment with sodium thiosulfate is effective&#46; We emphasize the need to maintain a multidisciplinary approach&#44; considering the comorbidities that occur in conjunction with this disease&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical responsibilities</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors confirm that they have obtained the informed consent of the patients and have complied with the ethical standards relevant to the conduct of this study&#46; This work has been consulted and approved by the Clinical Research Ethics Committee of the Hospital General de Granollers&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">The current study has not required funding for its completion&#46;</p></span></span>"
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Original language: English
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