Literature DB >> 17521035

Primary gastrointestinal lymphangiectasia presenting as cryptococcal meningitis.

Steven L Cole1, Dennis K Ledford, Richard F Lockey, Adel Daas, Jeffrey Kooper.   

Abstract

BACKGROUND: Opportunistic infections commonly occur in immunocompromised patients; however, it is unusual for an adult to present with a combined cellular and humoral immunodeficiency. Cryptococcal meningitis is a fatal condition if untreated and is usually found in patients with cellular immunodeficiency.
OBJECTIVE: To report the case of an adult patient with cryptococcal meningitis secondary to intestinal lymphangiectasia.
METHODS: A 59-year-old man was admitted to the hospital for disseminated cryptococcal meningitis and osteomyelitis. Laboratory evaluation, computed tomography, esophagogastroduodenoscopy, and biopsy were performed.
RESULTS: Laboratory evaluation revealed a lymphopenia, hypoalbuminemia, hypogammaglobulinemia, and negative human immunodeficiency virus test results by enzyme-linked immunosorbent assay and polymerase chain reaction. The complete blood cell count, urinalysis, serum and urine protein electrophoresis, and functional antibody responses to protein and polysaccharide antigens were normal. Results of computed tomography of the chest, abdomen, and pelvis were unremarkable. Multiple lymphangiectasias were visualized with esophagogastroduodenoscopy and confirmed by biopsy. The patient was treated with intravenous amphotericin B and flucytosine, and the meningitis resolved.
CONCLUSIONS: Based on a computerized search of the medical literature, this is the first description of cryptococcal meningitis secondary to intestinal lymphangiectasias. The combination of lymphopenia, hypogammaglobulinemia, and hypoalbuminemia should alert the clinician to the possibility of intestinal lymphangiectasias and the potential for immune dysfunction.

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Mesh:

Year:  2007        PMID: 17521035     DOI: 10.1016/S1081-1206(10)60765-X

Source DB:  PubMed          Journal:  Ann Allergy Asthma Immunol        ISSN: 1081-1206            Impact factor:   6.347


  6 in total

Review 1.  Primary intestinal lymphangiectasia: four case reports and a review of the literature.

Authors:  Jie Wen; Qingya Tang; Jiang Wu; Ying Wang; Wei Cai
Journal:  Dig Dis Sci       Date:  2010-03-03       Impact factor: 3.199

Review 2.  Primary intestinal lymphangiectasia: Minireview.

Authors:  Sachin B Ingle; Chitra R Hinge Ingle
Journal:  World J Clin Cases       Date:  2014-10-16       Impact factor: 1.337

Review 3.  Primary intestinal lymphangiectasia in an adult patient: A case report and review of literature.

Authors:  Rudolf Huber; Georg Semmler; Alexander Mayr; Felix Offner; Christian Datz
Journal:  World J Gastroenterol       Date:  2020-12-08       Impact factor: 5.742

4.  Disseminated Cryptococcal Disease in A Patient With Chronic Chylothorax and a Pleurovenous Catheter, a Case Report With Autopsy Findings.

Authors:  William Mundo; Amber Berning; Yiannis Koullias; Daniel B Chastain; Neil Stone; Carlos Franco-Paredes; Andrés F Henao-Martínez; Lilian Vargas Barahona
Journal:  Open Forum Infect Dis       Date:  2021-05-19       Impact factor: 3.835

5.  Cryptoccocal menigitis as a primary manifestation in a patient with intestinal lymphangictasia.

Authors:  Shaik Afshan Jabeen; Aruna Murthy; Rukmini Mridula Kandadai; A K Meena; Rupam Borgohain; Megha S Uppin
Journal:  Ann Indian Acad Neurol       Date:  2012-07       Impact factor: 1.383

Review 6.  Primary intestinal lymphangiectasia (Waldmann's disease).

Authors:  Stéphane Vignes; Jérôme Bellanger
Journal:  Orphanet J Rare Dis       Date:  2008-02-22       Impact factor: 4.123

  6 in total

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