RESUMEN
BACKGROUND: Vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome is caused by mutations in the ubiquitin-activating enzyme1 (UBA1) gene and characterised by an overlap between autoinflammatory and haematologic disorders. CASE PRESENTATION: We reported a case of a 67-year-Japanese man receiving peritoneal dialysis (PD) who had recurrent aseptic peritonitis caused by the VEXAS syndrome. He presented with unexplained fevers, headache, abdominal pain, conjunctival hyperaemia, ocular pain, auricular pain, arthralgia, and inflammatory skin lesions. Laboratory investigations showed high serum C-reactive protein concentration and increased cell count in PD effluent. He was treated with antibiotics for PD-related peritonitis, but this was unsuccessful. Fluorine-18-fluorodeoxyglucose (FDG) positron emission tomography/computed tomography images demonstrated intense FDG uptake in his left superficial temporal artery, nasal septum, and bilateral auricles. The working diagnosis was giant cell arteritis, and he was treated with oral prednisolone (PSL) 15 mg daily with good response. However, he was unable to taper the dose to less than 10 mg daily because his symptoms flared up. Since Tocilizumab was initiated, he could taper PSL dose to 2 mg daily. Sanger sequencing of his peripheral blood sample showed a mutation of the UBA1 gene (c.122 T > C; p.Met41Thr). We made a final diagnosis of VEXAS syndrome. He suffered from flare of VEXAS syndrome at PSL of 1 mg daily with his cloudy PD effluent. PSL dose of 11 mg daily relieved the symptom within a few days. CONCLUSIONS: It is crucial to recognise aseptic peritonitis as one of the symptoms of VEXAS syndrome and pay attention to the systemic findings in the patients.
Asunto(s)
Fluorodesoxiglucosa F18 , Síndromes Mielodisplásicos , Enfermedades Cutáneas Genéticas , Vacuolas , Humanos , Masculino , Dolor Abdominal , Mutación , Pacientes , AncianoRESUMEN
BACKGROUND: Oxaliplatin is an anticancer therapy for pancreatic, gastric, and colorectal cancers. It is also used in patients with carcinomas of unknown primary sites. Oxaliplatin is associated with less frequent renal dysfunction than other conventional platinum-based drugs such as cisplatin. Albeit, there have been several reports of acute kidney injury with frequent use. In all cases, renal dysfunction was temporary and did not require maintenance dialysis. There have been no previous reports of irreversible renal dysfunction after a single dose of oxaliplatin. CASE PRESENTATION: Previous reports of oxaliplatin-induced renal injury occurred after patients received multiples doses. In this study, a 75-year-old male with unknown primary cancer and underlying chronic kidney disease developed acute renal failure after receiving the first dose of oxaliplatin. Suspected of having drug-induced renal failure through an immunological mechanism, the patient was treated with steroids; however, treatment was ineffective. Renal biopsy ruled out interstitial nephritis and revealed acute tubular necrosis. Renal failure was irreversible, and the patient subsequently required maintenance hemodialysis. CONCLUSIONS: We provide the first report of pathology-confirmed acute tubular necrosis after the first dose of oxaliplatin which led to irreversible renal dysfunction and maintenance dialysis.
Asunto(s)
Lesión Renal Aguda , Fallo Renal Crónico , Necrosis Tubular Aguda , Neoplasias Primarias Desconocidas , Nefritis Intersticial , Masculino , Humanos , Anciano , Oxaliplatino/efectos adversos , Neoplasias Primarias Desconocidas/complicaciones , Diálisis Renal/efectos adversos , Fallo Renal Crónico/complicaciones , Necrosis Tubular Aguda/inducido químicamente , Necrosis Tubular Aguda/diagnóstico , Nefritis Intersticial/inducido químicamente , Nefritis Intersticial/diagnóstico , Nefritis Intersticial/complicaciones , Lesión Renal Aguda/inducido químicamente , Lesión Renal Aguda/diagnóstico , Lesión Renal Aguda/terapia , Biopsia/efectos adversos , NecrosisRESUMEN
Coronavirus disease 2019 (COVID-19) is a severe respiratory infection that can be fatal in unvaccinated individuals; however, acute kidney injury (AKI) is a rare adverse reaction to COVID-19 vaccination. AKI resulting from multiple conditions can have severe consequences, including end-stage renal failure, if not treated with immunosuppressive agents. However, acute tubular injury (ATI) as the sole cause of AKI has not been previously reported. Herein, we discuss an obese 54-year-old man with type 2 diabetes who received four COVID-19 vaccines; three from Pfizer and one from Moderna. Diabetic retinopathy, urinary protein, and occult blood were absent with no other underlying diseases. There was no history of COVID-19 infection. He was referred to our hospital 5 days after receiving the fourth Pfizer-BioNTech COVID-19 vaccine dose with stage 3 AKI. Urinary findings revealed new proteinuria and glomerular occult blood. Physical examination and infection testing were unremarkable. Steroids were introduced on admission for rapidly progressive glomerulonephritis. A renal biopsy performed on Day 2 revealed only ATI. Therefore, steroids were discontinued on Day 5, after which renal function recovered spontaneously, and urinalysis abnormalities disappeared. Renal function remained normal during follow-up. We report a case of AKI with severe renal dysfunction after COVID-19 vaccination, wherein renal biopsy effectively determined the disease status (ATI), which did not require immunosuppressive treatment.