Acute interstitial nephritis due to dolutegravir: The first case reported / Nefritis intersticial aguda por dolutegravir: primer caso notificado

Kidney disease is frequent in HIV-patients. We present a case of a 44-year-old woman, with known uncontrolled HIV infection and chronic kidney disease due to HIV-associated nephropathy. After starting dolutegravir, the patient developed eosinophilia and worsening kidney function. A kidney biopsy confirmed the diagnosis of acute interstitial nephritis. Given the time relation with dolutegravir introduction, it was deemed the culprit medication. Dolutegravir was stopped, and corticosteroids were initiated, with moderate improvement in renal function. To our knowledge, this is the first reported case of acute interstitial nephritis to dolutegravir, which should raise awareness of previously undocumented renal effects of antiretroviral therapy. (AU)

La enfermedad renal es frecuente en pacientes con VIH. Presentamos el caso de una mujer de 44 años, con infección por VIH no controlada conocida y enfermedad renal crónica por nefropatía asociada al VIH. Después de comenzar con dolutegravir, el paciente desarrolló eosinofilia y empeoramiento de la función renal. Una biopsia de riñón confirmó el diagnóstico de nefritis intersticial aguda. Dada la relación temporal con la introducción de dolutegravir, se consideró al medicamento el culpable. Se interrumpió el tratamiento con dolutegravir y se iniciaron corticosteroides, con una mejoría moderada de la función renal. Hasta donde sabemos, este es el primer caso notificado de nefritis intersticial aguda por dolutegravir, lo que debería crear conciencia sobre los efectos renales previamente indocumentados de la terapia antirretroviral. (AU)

Kidney Disease in Ankylosing Spondylitis: a case series and review of the literature / Doença Renal na Espondilite Anquilosante: uma série de casos e revisão da literatura

Abstract Background Kidney disease is a rare manifestation of ankylosing spondylitis (AS) and its pathological alterations remain poorly described. The aim of this study was to investigate the clinical presentation and pathological alterations on kidney biopsy of AS patients and review and discuss the current literature on the issue. Methods: We retrospectively studied the clinical presentation and kidney pathological alterations of 15 Caucasian AS patients submitted to kidney biopsy between October 1985 and March 2021. Results: Patients were predominantly male (66.7%) with median age at the time of kideney biopsy of 47 years [IQR 34 - 62]. Median serum creatinine at presentation was 1.3 mg/dL [IQR 0.9 - 3] and most patients also had either proteinuria (85.7%) and/or hematuria (42.8%). The most common indication for kidney biopsy was nephrotic syndrome (33.3%), followed by acute or rapidly progressive kidney injury (20%) and chronic kidney disease of unknown etiology (20%). Chronic interstitial nephritis (CIN) (n=3) and AA amyloidosis (n=3) were the most common diagnosis. Others included IgA nephropathy (IgAN) (n=2), focal segmental glomerulosclerosis (n=2), membranous nephropathy (n=1), and immune complex-mediated membranoproliferative glomerulonephritis (IC-MPGN)(n=1). Conclusions: We present one of the largest series of biopsy-proven kidney disease in Caucasian AS patients. We found a lower prevalence of IgAN than previously reported in Asian cohorts. We found a higher prevalence of CIN and a lower prevalence of AA amyloidosis than that described in previous series of Caucasian patients. We also present the first case of AS-associated IC-MPGN.

Resumo Antecedentes: A doença renal é uma manifestação rara de espondilite anquilosante (EA) e as suas alterações patológicas permanecem pouco descritas. O objetivo deste estudo foi investigar a apresentação clínica e alterações patológicas na biópsia renal de doentes com EA bem como rever e discutir a literatura atual sobre o assunto. Métodos: Estudamos retrospectivamente a apresentação clínica e alterações patológicas renais de 15 doentes caucasianos com EA submetidos a biópsia renal entre Outubro de 1985 e Março de 2021. Resultados: Os doentes eram predominantemente homens (66,7%) com idade mediana no momento da biópsia de 47 anos [IIQ 34 - 62]. A creatinina sérica mediana na apresentação foi de 1,3 mg/dL [IIQ 0,9 - 3] e a maioria dos pacientes apresentava também proteinúria (85,7%) e/ou hematúria (42,8%). A indicação mais comum para biópsia renal foi a síndrome nefrótica (33,3%), seguida de lesão renal aguda ou rapidamente progressiva (20%) e doença renal crónica de etiologia desconhecida (20%). A Nefrite intersticial crónica (NIC) (n=3) e a amiloidose AA (n=3) foram os diagnósticos mais comuns. Outros incluíram nefropatia por IgA (NIgA) (n=2), glomeruloesclerose segmentar focal (n=2), nefropatia membranosa (n=1) e glomerulonefrite membranoproliferativa mediada por imunocomplexos (GNMP-IC) (n=1). Conclusões: Apresentamos uma das maiores séries de doenças renais comprovadas por biópsia em doentes caucasianos com EA. Encontramos uma prevalência de NIgA menor do que a relatada anteriormente em coortes asiáticas. Encontramos uma maior prevalência de NIC e uma prevalência menor de amiloidose AA do que a descrita em séries anteriores de pacientes caucasianos. Também apresentamos o primeiro caso de GNMP-IC associada à EA.

Urinary Sediment Microscopy and Correlations with Kidney Biopsy: Red Flags Not To Be Missed.

BACKGROUND: Urinary sediment is a noninvasive laboratory test that can be performed by an automated analyzer or manually by trained personnel. Manual examination remains the diagnostic standard because it excels at differentiating isomorphic from dysmorphic red blood cells and identifying other urinary particles such as renal tubular epithelial cells (RTECs), lipids, crystals, and the composition of casts. This study aimed to investigate the prevalence of a complete profile of urinary sediment particles and its associations with histologic lesions on kidney biopsy, regardless of diagnosis. METHODS: This was a single-center, observational retrospective study of 131 patients who had contemporary manual urinary sediment evaluation and kidney biopsy. A comprehensive set of urinary particles and histologic lesions were quantified, and their associations were analyzed. RESULTS: In our samples, we found an elevated frequency of findings suggestive of proliferative kidney disease and a low frequency of particles evoking urologic damage. The association of histologic lesions and urinary particles was explored with a multivariate model. We identified urinary sediment characteristics that independently correlated with the presence of some histologic lesions: urinary lipids with mesangial expansion (OR=2.86; 95% confidence interval [95% CI], 1.3 to 6.3), mesangial hypercellularity (OR=2.44; 95% CI, 1.06 to 5.58), and wire loops and/or hyaline deposits (OR=2.89; 95% CI, 1.13 to 7.73); Urinary renal tubular epithelial cells with endocapillary hypercellularity (OR=3.17; 95% CI, 1.36 to 7.39), neutrophils and/or karyorrhexis (OR=4.51; 95% CI, 1.61 to 12.61), fibrinoid necrosis (OR=4.35; 95% CI, 1.48 to 12.74), cellular/fibrocellular crescents (OR=5.27; 95% CI, 1.95 to 14.26), and acute tubular necrosis (OR=2.31; 95% CI, 1.08 to 4.97). CONCLUSIONS: In a population of patients submitted to kidney biopsy, we found that the presence of some urinary particles (renal tubular epithelial cells, lipids, and dysmorphic erythrocytes), which are seldom reported by automated analyzers, is associated with active proliferative histologic lesions. In this regard, manual urinary sediment evaluation may help to shape the indications for performing a kidney biopsy.

Kidney Disease in Ankylosing Spondylitis: a case series and review of the literature.

BACKGROUND: Kidney disease is a rare manifestation of ankylosing spondylitis (AS) and its pathological alterations remain poorly described. The aim of this study was to investigate the clinical presentation and pathological alterations on kidney biopsy of AS patients and review and discuss the current literature on the issue. METHODS: We retrospectively studied the clinical presentation and kidney pathological alterations of 15 Caucasian AS patients submitted to kidney biopsy between October 1985 and March 2021. RESULTS: Patients were predominantly male (66.7%) with median age at the time of kideney biopsy of 47 years [IQR 34 - 62]. Median serum creatinine at presentation was 1.3 mg/dL [IQR 0.9 - 3] and most patients also had either proteinuria (85.7%) and/or hematuria (42.8%). The most common indication for kidney biopsy was nephrotic syndrome (33.3%), followed by acute or rapidly progressive kidney injury (20%) and chronic kidney disease of unknown etiology (20%). Chronic interstitial nephritis (CIN) (n=3) and AA amyloidosis (n=3) were the most common diagnosis. Others included IgA nephropathy (IgAN) (n=2), focal segmental glomerulosclerosis (n=2), membranous nephropathy (n=1), and immune complex-mediated membranoproliferative glomerulonephritis (IC-MPGN)(n=1). CONCLUSIONS: We present one of the largest series of biopsy-proven kidney disease in Caucasian AS patients. We found a lower prevalence of IgAN than previously reported in Asian cohorts. We found a higher prevalence of CIN and a lower prevalence of AA amyloidosis than that described in previous series of Caucasian patients. We also present the first case of AS-associated IC-MPGN.

Acute interstitial nephritis due to dolutegravir: The first case reported.

Kidney disease is frequent in HIV-patients. We present a case of a 44-year-old woman, with known uncontrolled HIV infection and chronic kidney disease due to HIV-associated nephropathy. After starting dolutegravir, the patient developed eosinophilia and worsening kidney function. A kidney biopsy confirmed the diagnosis of acute interstitial nephritis. Given the time relation with dolutegravir introduction, it was deemed the culprit medication. Dolutegravir was stopped, and corticosteroids were initiated, with moderate improvement in renal function. To our knowledge, this is the first reported case of acute interstitial nephritis to dolutegravir, which should raise awareness of previously undocumented renal effects of antiretroviral therapy.

[A rare presentation of kidney allograft intolerance syndrome: Bullous pemphigoid]. / Une présentation rare du syndrome d'intolérance au greffon renal : la pemphigoïde bulleuse.

Bullous pemphigoid is an autoimmune bullous cutaneous disease. We report the case of a 60 year-old male patient whose kidney allograft failed and was on hemodialysis for the previous 16 months. After tapering immunosuppressive medication, he presented simultaneous bullous eruption and kidney allograft intolerance syndrome. Investigation showed a positive BP180 anti-basement membrane zone antibody and skin biopsy was consistent with bullous pemphigoid. The patient was treated with corticotherapy and bullous pemphigoid resolved. The development of new onset diabetes and concerns over long term immunosuppression, halted the decision to continue corticotherapy and the patient underwent graft nephrectomy, with resolution of the kidney allograft intolerance syndrome without recurrence of the bullous disease. The occurrence of bullous pemphigoid in patients with failed renal allograft is rare, with only eleven cases reported in literature. This case illustrates how graft nephrectomy can provide a definitive cure to bullous pemphigoid in this setting.

Etanercept in the treatment of ankylosing spondylitis and nephrotic syndrome in the context of AA amyloidosis: A 48-month follow-up / Etanercept en el tratamiento de la espondilitis anquilosante y el síndrome nefrótico en el contexto de la amiloidosis aa: un seguimiento de 48 meses

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Uncracking a Case.

Nutcracker phenomenon (NCP) refers to compression of the left renal vein (LRV), most frequently between the aorta and the superior mesenteric artery, with impaired blood outflow often accompanied by distention of the distal portion of the vein. The nutcracker syndrome (NCS) is the terminology used when the nutcracker phenomenon is accompanied by a complex of symptoms such as pain (abdominal, flank, and pelvic), hematuria and orthostatic proteinuria. Diagnosis can be made with Doppler ultrasound (DUS), computed tomography (CT), magnetic resonance imaging (MRI), and venography. We describe a case of a young female adult who was identified with NCP by DUS, after a first CT had found no abnormalities. She presented with flank pain and severe hematuria causing a drastic decrease in hemoglobin levels. The management of NCS depends upon the clinical presentation and the severity of the LRV hypertension. The treatment options range from conservative to nephrectomy. Treatment decision should be based on the age of patients, severity of symptoms, and their expected reversibility. This case describes an underreported disorder that presents with non-specific symptoms, demonstrating the difficulties of the diagnostic approach of NCS, as well as the challenges in the appropriate management, given the lack of standardized treatment.

Rare diagnosis in a patient with diabetes with nephrotic proteinuria.

We report a 63-year-old man with well-controlled type 2 diabetes mellitus and hypertension, who presented with new onset nephrotic proteinuria and rapid deterioration in renal function. The atypical clinical presentation prompted us to consider a non-diabetic and non-hypertensive cause and to perform a renal biopsy. A diagnosis of fibrillarglomerulonephritis (FGn) was made based on electronic microscopy. Proteinuria remained in nephrotic range despite treatment with prednisolone, and renal function deteriorated. We suggest that other causes of proteinuria should be considered in patients with diabetes who present with the nephrotic syndrome when there is no other evidence of microvascular disease. We review the spectrum of fibrillar glomerulopathies including FGn, primary and secondary amyloidosis and immunotactoid glomerulonephritis.

MPO-ANCA-associated necrotizing glomerulonephritis in rheumatoid arthritis; a case report and review of literature.

BACKGROUND: Renal involvement in rheumatoid arthritis (RA) is common and has a negative impact on patient survival. Only few cases have been reported of necrotizing glomerulonephritis (GN) associated with myeloperoxidase anti-neutrophil cytoplasmic antibody (MPO-ANCA) in patients with RA. CASE PRESENTATION: We report a patient with RA who developed a necrotizing GN associated with ANCA-MPO, treated with rituximab (RTX). A 55-year-old man with a 27-year history of RA under secukinumab was referred to our nephrology clinic with worsening renal function associated with microhematuria and proteinuria. Our laboratory evaluation showed hypocomplementemia and positive titers for MPO-ANCA (615 U/mL). A renal biopsy demonstrated pauci-immune necrotizing GN. The patient was treated with 3 consecutive pulses of methylprednisolone followed by oral prednisolone (1 mg/Kg) and rituximab (1000 mg, repeated 14 days later). After a 10-month follow-up, the arthritis remains well-controlled, renal function stabilized, proteinuria improved and MPO-ANCA titer normalized (6.3 U/mL). CONCLUSIONS: Necrotizing GN is a rare but a serious condition and an early diagnosis is essential to treatment. This is the first case of necrotizing GN (without extra-renal manifestations of vasculitis) in a patient with active RA, successfully treated with RTX.

Anticoagulant-related nephropathy in a patient with IgA nephropathy.

Anticoagulant-related nephropathy is a type of acute kidney injury caused by overcoagulation. We describe a case of an 84-year-old man with arterial hypertension, coronary heart disease and atrial fibrillation treated with acenocoumarol, who presented with haematoproteinuria and acute kidney injury during a phase of excessive anticoagulation. In addition to IgA nephropathy, renal biopsy also revealed acute tubular necrosis, red blood cell casts and positive iron staining in tubular cells. After this acute episode, renal function improved and proteinuria decreased below the nephrotic range.