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Case Report: Capillary Leak Syndrome With Kidney Transplant Failure Following Autologous Mesenchymal Stem Cell Therapy.

ABSTRACT: Mesenchymal stem cells (MSCs) have attracted great interest in the field of kidney transplantation due to their immunomodulatory and reparative properties. In registered clinical trials, MSCs have been used before, at the time of, or early after transplantation and have been reported to be well-tolerated with no serious safety concerns. No results are available on the use of MSCs in the late post-transplant period. Here, we present a case report of a severe systemic complication mimicking capillary leak syndrome with ultimate kidney transplant failure after autologous transplantation of MSCs used as rescue treatment of late antibody-mediated kidney allograft rejection.

SUBMITTER: Veceric-Haler Z

PROVIDER: S-EPMC8334176 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Systemic leak capillary syndrome with myocardial involvement and cardiogenic shock: a case report.

Project description:BackgroundSystemic capillary leak syndrome (SCLS) is a potentially fatal disorder characterized by relapses of hypovolemic shock episodes.Case summaryWe present a case of a 58-year-old man who presented to the Emergency Department with a history of recurrent episodes of syncope in the last hours. A few days before medical contact the patient complained of sore throat, fever, and flu-like symptoms. He was initially admitted with a diagnosis of suspected myopericarditis. Forty-eight hours later, the haemodynamic status suddenly deteriorated to a mixed cardiogenic and shock; an endomyocardial biopsy showed localized inflammatory infiltrates and areas of necrosis of cardiomyocytes with positive viral search for parvovirus B19 (PVB19), therefore the patient was treated with methylprednisolone pulses. Based on the concurrent presence of the typical triad of hypotension, hypoalbuminaemia, and haemoconcentration we suspected systemic leak capillary syndrome potentially triggered by the PVB19 infection with acute myocarditis. The clinical conditions further deteriorated with rhabdomyolysis and acute kidney injury: we started continuous veno-venous haemofiltration adding a cytokines adsorber. In the following hours, we observed a significant clinical improvement. The patient was discharged 1 month later and 5 months after discharge he experienced a new attack of SCLS, this time without myocardial involvement and with prompt symptoms resolution.ConclusionSystemic capillary leak syndrome is a potentially fatal disorder: early recognition of this entity and prompt initiation of supportive therapy are warranted, therefore, it is paramount that an emergency physician thinks of SCLS in patients with signs of cardiogenic shock and the classical triad of hypotension, hypoalbuminia, and haemoconcentration.

| S-EPMC9258032 | biostudies-literature

A case report of capillary leak syndrome with recurrent pericardial and pleural effusions.

Project description:BackgroundCapillary leak syndrome (CLS) is a rare connective tissue disease, triggered by the leak of serous fluid into the interstitial spaces, characterized by a hallmark of oedema and effusions in confined spaces. The limiting factor in CLS management appears to be its diagnosis rather than treatment, which is usually to contain the disease progression rather than a cure.Case summaryWe report a case of a 51-year-old woman with recurrent life-threatening presentations of pericardial effusions, pleural effusions, and generalized swelling of face and extremities. The only notable past medical history was of Type 1 diabetes. Numerous investigations did not lead to specific disease accounting for pericardial effusions and pleural effusions. Eventually, the diagnosis of CLS was made based on hypovolaemic shock, hypoalbuminaemia, and haemoconcentration without the presence of albuminuria. She was managed with steroids to reduce system inflammation and later with immunoglobulins and tumour necrosis factor to contain the disease process. Since her diagnosis and subsequent appropriate management, she has not had further admissions with cardiac tamponade 16 months of follow-up.DiscussionThe diagnosis of CLS is difficult to make unless there is a high degree of suspicion and until other causes have been ruled out. It remains a challenging condition to manage as the treatment options are limited and patients recurrently present with emergencies until the correct diagnosis is made and the optimal treatment is provided.

| S-EPMC7180544 | biostudies-literature

Kidney transplant tolerance associated with remote autologous mesenchymal stromal cell administration.

Project description:Here we report the case of successful immune tolerance induction in a living-donor kidney transplant recipient remotely treated with autologous bone marrow-derived mesenchymal stromal cells (MSC). This case report, which to the best of our knowledge is the first in the world in this setting, provides evidence that the modulation of the host immune system with MSC can enable the safe withdrawal of maintenance immunosuppressive drugs while preserving optimal long-term kidney allograft function.

| S-EPMC7103624 | biostudies-literature

Case report: percutaneous closure of residual leak following left atrial appendage occlusion.

Project description:BackgroundTranscatheter left atrial appendage occlusion (LAAO) using Watchman device has been demonstrated to be efficacious in decreasing stroke risk in patients with atrial fibrillation who are not suitable for long-term anticoagulation. Residual leaks are frequently encountered following LAAO procedures and their clinical implications and optimal management remain controversial.Case summaryIn this report, we describe a case of peri-Watchman device leak treated successfully with percutaneous device closure using an Amplatzer Vascular Plug II device.DiscussionThe clinical implications of peri-device leaks remain controversial with general consensus to continue anticoagulation along with serial imaging for larger leaks (>5 mm). As an alternative strategy, percutaneous closure of these leaks has been attempted in hope of avoiding anticoagulation and minimizing the risk of stroke and should be studied further.

| S-EPMC7501943 | biostudies-literature

Capillary leak-syndrome triggered by Maripa virus in French Guiana: case report and implication for pathogenesis.

Project description:BackgroundWe report hereby a severe case of Hantavirus Pulmonary Syndrome" (HPS) induced by Maripa virus in French Guiana and describe the mechanism of severity of the human disease.Case presentationA 47-year- old patient started presenting a prodromic period with fever, dyspnea, cough and head ache. This clinical presentation was followed by a rapid respiratory, hemodynamic and renal failure leading to admission in the ICU. Biological exams revealed an increased haematocrit level with a paradoxical low protein level. Echocardiographic and hemodynamic monitoring showed a normal left ventricular function with low filling pressures, an elevated extravascular lung water index and pulmonary vascular permeability index. These findings were compatible with a capillary leak-syndrome (CLS).ConclusionsThe severity of HPS caused by the virus Maripa in French Guiana can be explained by the tropism of hantavirus for the microvascular endothelial cell leading to a CLS.

| S-EPMC6420753 | biostudies-literature

Capillary leak syndrome presenting as refractory ascites in a patient with lymphoma: A rare case report.

Project description:Capillary leak syndrome (CLS) is a serious disorder characterized by hypotension and refractory systemic oedema. CLS with marked ascites rather than systemic oedema is rare and prone to misdiagnosis and delayed treatment. We report here a case of marked ascites in an elderly male patient with hepatitis B virus reactivation. Following investigations to exclude common diseases that may have accounted for diffuse oedema and hypercoagulable state, anti-cirrhosis therapy failed and severe refractory shock developed 48 hours after admission. The patient developed mild pleural effusions followed by swelling of the face, neck, and extremities. A high cytokine concentration gradient was detected between serum and ascites. Peritoneal biopsy showed lymphoma cells. The final diagnosis was lymphoma recurrence complicated with CLS. Our case suggests that cytokine detection in serum and ascitic fluid may be helpful in the differential diagnosis of CLS. In similar cases, decisive intervention, such as, hemodiafiltration, should be implemented to lessen the likelihood of serious complications.

| S-EPMC10228172 | biostudies-literature

Efficacy and Immunomodulating Properties of Eltrombopag in Aplastic Anemia following Autologous Stem Cell Transplant: Case Report and Review of the Literature.

Project description:Thrombopoietin receptor agonists (TPO-RA) are currently indicated for the treatment of chronic immune thrombocytopenia and relapsed refractory aplastic anemia. However, the off-label use of these drugs is more and more frequent, including in the setting of aplasia secondary to chemotherapy and hemopoietic stem cell transplant (SCT). Growing evidence suggests that mechanisms of action of TPO-RA go beyond the TPO-receptor stimulation and point at the immunomodulating properties of these drugs. Here, we present a case of prolonged bone marrow aplasia secondary to autologous SCT treated with eltrombopag. We describe the clinical efficacy and the immunomodulating effect of this drug on inflammatory cytokine profile and bone marrow histology. Furthermore, we provide a review of the most recent literature highlighting the efficacy and safety of TPO-RA after SCT and chemotherapy for hematologic conditions.

| S-EPMC9032708 | biostudies-literature

A case of cryopyrin-associated periodic syndrome with kidney transplant failure.

Project description:The cryopyrin-associated periodic syndrome (CAPS) is an autosomal dominant autoinflammatory disease characterized by fever, skin rash, and joint involvement with acute inflammatory response. The genetic defect involves the NLRP3 gene that encodes cryopyrin and leads to an abnormal production of interleukin-1 (IL-1). Therefore, anti-IL-1 treatment represents an effective therapy. One of the most severe manifestations of the disease is secondary amyloidosis that causes renal failure. We present a patient with CAPS who underwent renal transplantation for renal insufficiency caused by amyloidosis. The function of the transplanted kidney deteriorated because of the late administration of IL-1 receptor antagonist, anakinra. This case may indicate the importance of early initiation of anti-IL-1 treatment in CAPS patients who have undergone kidney transplantation.

| S-EPMC5411619 | biostudies-literature

Continuous renal replacement therapy rescued life-threatening capillary leak syndrome in an extremely-low-birth-weight premature: a case report.

Project description:BackgroundCapillary leak syndrome (CLS) is a rare disease characterized by profound vascular leakage and presents as a classic triad of hypotension, hypoalbuminemia and hemoconcentration. Severe CLS is mostly induced by sepsis and generally life-threatening in newborns, especially in premature infants. Continuous renal replacement therapy (CRRT) plays an important role of supportive treatment for severe CLS. Unfortunately, CRRT in preterm infants has rarely been well defined.Case presentationWe report the case of a 11-day-old girl with CLS caused by sepsis, who was delivered by spontaneous vaginal delivery (SVD) at gestational age of 25 weeks and 4 days, and a birth weight of 0.89 Kilograms(kg). The infant received powerful management consisting of united antibiotics, mechanical ventilation, intravenous albumin and hydroxyethyl starch infusion, vasoactive agents, small doses of glucocorticoids and other supportive treatments. However, the condition rapidly worsened with systemic edema, hypotension, pulmonary exudation, hypoxemia and anuria in about 40 h. Finally, we made great efforts to perform CRRT for her. Fortunately, the condition improved after 82 h' CRRT, and the newborn was rescued and gradually recovered.ConclusionCRRT is an effective rescue therapeutic option for severe CLS and can be successfully applied even in extremely-low-birth-weight premature.

| S-EPMC8157435 | biostudies-literature

Flood syndrome following right-sided heart failure: a case report.

Project description:Flood syndrome is a rare condition, where a patient with ascites suffers sudden umbilical hernia rupture and a subsequent drainage of ascitic fluid from the abdominal cavity. The cause of ascites is usually liver cirrhosis. Complications associated with cirrhosis of the liver make Flood syndrome difficult to manage. In these cases, conservative management has been associated with high morbidity. We describe, to our knowledge, for the first time a patient with Flood syndrome caused by right-sided heart failure. We also show that conservative treatment gives great initial outcome and is a viable option for this type of Flood syndrome.

| S-EPMC8790074 | biostudies-literature

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