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Abstract Number: 2123

Protein-Losing Enteropathy in Patients with Systemic Autoimmune Diseases: Characterization of 263 Cases (GEAS-SEMI Spanish Cohort)

Alejandra Flores-Chavez1,2,3, Soledad Retamozo4,5,6, Angel Robles7, Guadalupe Fraile Rodriguez8, Sofia Arteaga9,10, Celeste Galceran-Chaves11, Roberto Pérez-Alvarez12, Marta Pérez de Lis12, Belchin Kostov13, Manuel Ramos-Casals14,15 and Pilar Brito-Zerón14,16, 1Department of Autoimmune Diseases, ICMiD, Hospital Clínic Barcelona, Spain., Barcelona, Spain, 2Unidad de Investigación Biomédica 02 (Unidad de Investigación en Epidemiología Clínica), UMAE, Hospital de Especialidades, Centro Médico Nacional de Occidente, Instituto Mexicano del Seguro Social, Jalisco, Mexico, 3Programa de Doctorado en Ciencias Médicas, Centro Universitario de Investigaciones Biomédicas (CUIB), Universidad de Colima, Colima, Mexico, Mexico, Mexico, 4Instituto de Investigaciones en Ciencias de la Salud, Universidad Nacional de Córdoba, Consejo Nacional de Investigaciones Científicas y Técnicas (INICSA-UNC-CONICET), Cordoba, Argentina, 5Laboratory of Systemic Autoimmune Diseases “Josep Font”, CELLEX, Institut d’Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS), Department of Systemic Autoimmune Diseases, ICMID, Hospital Clinic, Barcelona, Barcelona, Spain, 6Rheumatology Unit, Hospital Privado Universitario de Córdoba, Institute University of Biomedical Sciences University of Córdoba (IUCBC), Cordoba, Argentina, 7Department of Internal Medicine, Hospital La Paz, Madrid, Madrid, Spain, 8Internal Medicine, Hospital Ramón y Cajal, Madrid, Madrid, Spain, 9Department of Autoimmune Diseases, ICMiD, Hospital Clínic Barcelona, Barcelona, Spain, 10Universidad de Antioquía, Medellín, Colombia, Medellin, Colombia, 11Neuroscience Clinical Institute, Hospital Clínic, Barcelona, Barcelona, Spain, 12Department of Internal Medicine, Hospital Alvaro Cunqueiro, Vigo, Vigo, Spain, 13Primary Care Research Group, IDIBAPS, Centre d’Assistència Primària ABS Les Corts, CAPSE, Barcelona, Barcelona, Spain, 14Laboratory of Systemic Autoimmune Diseases “Josep Font”, CELLEX, Institut d’Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS), Department of Systemic Autoimmune Diseases, ICMID, Hospital Clinic, Barcelona, Spain, Barcelona, Spain, 15Department of Medicine, University of Barcelona, Barcelona, Spain., Barcelona, Spain, 16Autoimmune Diseases Unit, Department of Medicine, Hospital CIMA- Sanitas, Barcelona., Bacelona, Spain

Meeting: 2017 ACR/ARHP Annual Meeting

Date of first publication: September 18, 2017

Keywords: autoimmune diseases and sarcoidosis, Lupus, Sjogren's syndrome

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Session Information

Date: Tuesday, November 7, 2017

Session Title: Miscellaneous Rheumatic and Inflammatory Diseases Poster II

Session Type: ACR Poster Session C

Session Time: 9:00AM-11:00AM

Background/Purpose: Protein-losing enteropathy (PLE) is a rare condition characterized by a loss of serum protein into the gastrointestinal tract resulting in hypoproteinemia, which can be complicated by edema, ascites, pleural and pericardial effusions, and malnutrition. Although rare, PLE may complicate a variety of diseases, most commonly cardiac or gastrointestinal conditions, and less infrequently, systemic autoimmune diseases (SAD).

Methods: In January 2016, we created a retrospective multicenter collection of cases of PLE reported in patients with SAD through the Spanish Autoimmune Diseases Study Group (GEAS-SEMI), and we carried out a systematic literature review. PLE was clinically diagnosed in patients presenting with hypoalbuminemia due to gastrointestinal loss after discarding urinary protein loss and reduced protein synthesis (malnutrition, liver disease, etc). Patients in whom evidence of protein leakage from the gastrointestinal tract as detected by 99 m Tc-HSA scintigraphy were classified as definite PLE, and those patients in which this examination was not performed were classified as probable PLE.

Results: A total of 263 patients with SAD-related PLE (6 patients from the Registry and 210 from the literature review) were included (80% were women, with a mean age at diagnosis of disease of 41.1 years, range 16-79 years); 226 (86%) of cases were reported from Asian countries (64% from China), 24 from European countries and 13 from America (7 from US). The main underlying SAD was systemic lupus erythematosus (SLE) in 202 (77%) cases, followed by Sjögren syndrome in 18 (7%) and sarcoidosis in 15 (6%). The main signs and symptoms at presentation were peripheral edema in 192 (85%) patients, pleural effusion in 129 (57%), ascites in 120 (53%) and diarrhea in 96 (43%) patients. Internal organ involvement CNS (in 8 (16%), pulmonary in 23 (46%) and renal in 15 (30%). The main analytical markers consisted of hypercholesterolemia (90%), hypertriglyceridemia (85%) and positive alpha1-antitrypsin in stool (68%). The main gastrointestinal endoscopic finding consisted of edematous mucosa either in upper endoscopy (67/129, 52%) and lower endoscopy (74/139, 53%). One hundred and eighty-two (69%) patients were classified as definite PLE and 81 (31%) were classified as probable PLE. Therapies included corticosteroids in 162 patients and immunosuppressive agents in 171 (mainly cyclophosphamide and azathioprine); biological agents were used in 5 patients (rituximab in 4, infliximab in 1). After a mean follow-up of 15 months, 77% of patients had a complete response, 6% developed a disease relapse, and 4% died.

Conclusion: A very specific epidemiological and clinical profile was observed for protein-losing enteropathy associated with systemic autoimmune diseases, with nearly 90% of cases reported from Asian countries (two out of three cases from China), and nearly 80% reported in patients with underlying SLE. Patients were overwhelmingly treated with corticosteroids and immunosuppressive agents with a complete response of nearly 80% of cases and a global mortality of less than 5%.


Disclosure: A. Flores-Chavez, None; S. Retamozo, None; A. Robles, None; G. Fraile Rodriguez, None; S. Arteaga, None; C. Galceran-Chaves, None; R. Pérez-Alvarez, None; M. Pérez de Lis, None; B. Kostov, None; M. Ramos-Casals, None; P. Brito-Zerón, None.

To cite this abstract in AMA style:

Flores-Chavez A, Retamozo S, Robles A, Fraile Rodriguez G, Arteaga S, Galceran-Chaves C, Pérez-Alvarez R, Pérez de Lis M, Kostov B, Ramos-Casals M, Brito-Zerón P. Protein-Losing Enteropathy in Patients with Systemic Autoimmune Diseases: Characterization of 263 Cases (GEAS-SEMI Spanish Cohort) [abstract]. Arthritis Rheumatol. 2017; 69 (suppl 10). https://acrabstracts.org/abstract/protein-losing-enteropathy-in-patients-with-systemic-autoimmune-diseases-characterization-of-263-cases-geas-semi-spanish-cohort/. Accessed April 13, 2021.
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