Literature DB >> 33011939

Clinical Spectrum of Ras-Associated Autoimmune Leukoproliferative Disorder (RALD).

Quentin Neven1, Cécile Boulanger2, Annelyse Bruwier3, Maëlle de Ville de Goyet2, Isabelle Meyts4,5, Leen Moens4, An Van Damme2, Bénédicte Brichard2.   

Abstract

Ras-associated autoimmune leukoproliferative disorder (RALD) is a clinical entity initially identified in patients evaluated for an autoimmune lymphoproliferative syndrome (ALPS)-like phenotype. It remains a matter of debate whether RALD is a chronic and benign lymphoproliferative disorder or a pre-malignant condition. We report the case of a 7-year-old girl diagnosed with RALD due to somatic KRAS mutation who progressed to a juvenile myelomonocytic leukemia phenotype and finally evolved into acute myeloid leukemia. The case report prompted a literature review by a search for all RALD cases published in PubMed and Embase. We identified 27 patients with RALD. The male-to-female ratio was 1:1 and median age at disease onset was 2 years (range 3 months-36 years). Sixteen patients (59%) harbored somatic mutations in KRAS and 11 patients (41%) somatic mutations in NRAS. The most common features were splenomegaly (26/27 patients), autoimmune cytopenia (15/16 patients), monocytosis (18/24 patients), pericarditis (6 patients), and skin involvement (4 patients). Two patients went on to develop a hematopoietic malignancy. In summary, the current case documents an additional warning about the long-term risk of malignancy in RALD.

Entities:  

Keywords:  Autoimmunity; KRAS; NRAS; Ras-associated autoimmune leukoproliferative disorder; malignancy

Mesh:

Substances:

Year:  2020        PMID: 33011939     DOI: 10.1007/s10875-020-00883-7

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  1 in total

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Journal:  Blood       Date:  1997-05-15       Impact factor: 22.113

  1 in total
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Review 4.  Gain-of-function defects in toll-like receptor 8 shed light on the interface between immune system and bone marrow failure disorders.

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Review 5.  Lymphadenopathy at the crossroad between immunodeficiency and autoinflammation: An intriguing challenge.

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Journal:  Clin Exp Immunol       Date:  2021-06-20       Impact factor: 4.330

  5 in total

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