Coupled Plasma Filtration Adsorption for Treatment of Capillary Leak Syndrome Superimposed to Acute Generalized Exanthematous Pustolosis: A Case Report

Abstract

AbstarctCoupled plasma filtration adsorption (CPFA) is an extracorporeal supportive therapy based on nonspecific adsorption of pro-and anti-inflammatory mediators combined with continuous renal replacement therapy. The main field of CPFA application is septic shock, and there are limited data about its efficacy in the treatment of other acute conditions characterized by a dysregulation in immune homeostasis. Capillary leak syndrome (CLS) defines a life-threatening condition sustained by hypercytokinemia and characterized by abrupt onset of increased capillary permeability leading to severe generalized edema and hypovolemic shock refractory to fluid administration. Therapy for CLS is not specific and, at present time, it consists in the use of steroids or intravenous immunoglobulins. We present the case of a 34-yearold woman who developed CLS superimposed to acute generalized exanthematous pustulosis after initiating therapy with hydroxychloroquine for undifferentiated connective tissue disease. CLS did not respond to steroids and intravenous immunoglobulins, while it was successfully treated with CPFA. This observation supports the possible role of CPFA in restoring a proper immunologic homeostasis not only in sepsis but also in other devastating conditions sustained by hypercytokinemia.

Hydroxychloroquine‐induced acute generalized exanthematous pustulosis: a series of seven patients and review of the literature

“…For example, the delay between drug exposure and symptom onset varied from 1 day to 3 weeks in diltiazem‐induced AGEP 10 , and from 1 day to 44 days in terbinafine‐induced AGEP 11 . While, in previously reported HCQ‐induced AGEP cases, the duration of exposure to HCQ prior to symptom onset ranged from 2 12 to 30 days 13,14 . In our patients, except for the lichen planus case who developed AGEP symptoms 122 days after onset of treatment, most cases of our series developed symptoms between 15 and 47 days, which is similar to the literature.…”

Clinical use of plasmafiltration coupled with adsorption

“…Recently, the first case of CPFA was reported in a patient with severe and refractory capillary leakage associated with acute generalized exanthematous pustulosis, and good results were obtained. [56] Early identification of capillary leaks and installation of CPFA may be a useful tool to avoid overuse of leaks. In these cases, the main purpose is to seek immune regulation under clinical conditions.…”

Particulars of the comprehensive treatment of the patients with the new coronavirus infection (COVID-19): guidelines on the in-patient management

“…Характеристики: высокая проницаемость пор с заданным cut-off на уровне до 40 кДа; повышенный клиренс молекул средней массы, цитокинов и других медиаторов воспаления; кривая коэффициента просеивания мембраны практически идентична кривой просеивания клубочковой мембраны почки. [38][39][40][41].…”