Literature DB >> 23588339

Tubulointerstitial nephritis in a patient with probable autoimmune lymphoproliferative syndrome.

Mia Glerup1, Troels Herlin, Søren Rittig, Kirsten Grønbæk, Marianne Hokland, Henrik Hasle.   

Abstract

Autoimmune lymphoproliferative syndrome (ALPS) is caused by a nonmalignant defective Fas-mediated apoptosis. The main clinical manifestations are chronic lymphadenopathy, splenomegaly, and autoimmune cytopenia. Most patients with ALPS have a FAS germline mutation. ALPS has occasionally been associated with glomerulonephritis and we present the first report of tubulointerstitial nephritis associated with probable ALPS. A 5-year-old girl presented with fever, vomiting, hypertension, and azotemia. No autoantibodies, viral, or streptococcal antibodies were detected. A renal biopsy showed small-vessel vasculitis with normal glomeruli and inflammation in the interstitium. The patient responded to prednisolone treatment and obtained a full renal recovery. Symptoms of connective tissue disorder supervened and after the development of more pronounced splenomegaly, a diagnosis of ALPS was confirmed.

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Year:  2013        PMID: 23588339     DOI: 10.1097/MPH.0b013e31828ac9fe

Source DB:  PubMed          Journal:  J Pediatr Hematol Oncol        ISSN: 1077-4114            Impact factor:   1.289


  2 in total

1.  Lymphadenopathy, splenomegaly, intermittent neutropenia, and acute kidney injury: Answers.

Authors:  Katie Sullivan; Rose Chami; Rachel Pearl
Journal:  Pediatr Nephrol       Date:  2019-08-29       Impact factor: 3.714

Review 2.  Rheumatologic manifestations of primary immunodeficiency diseases.

Authors:  V R Dimitriades; R Sorensen
Journal:  Clin Rheumatol       Date:  2016-03-14       Impact factor: 2.980

  2 in total

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