How to treat an extensive form of primary intestinal lymphangiectasia?
Identifieur interne : 000166 ( Pmc/Curation ); précédent : 000165; suivant : 000167How to treat an extensive form of primary intestinal lymphangiectasia?
Auteurs : Rosana Troskot ; Dragan Jur I ; Ante Bili ; Marija Gomer I Pal I ; Stanko Težak ; Ivana BrajkoviSource :
- World Journal of Gastroenterology : WJG [ 1007-9327 ] ; 2015.
Abstract
We report a case of a 42-year-old man with a rare disorder known as primary intestinal lymphangiectasia, which is characterized by dilated intestinal lymphatics that lead to the development of protein-losing enteropathy. The patient presented with a grand mal seizure caused by malabsorption-derived electrolytes and a protein disorder. Signs of the disease, including chronic diarrhea and peripheral edema, manifested 10 years ago, but a diagnosis was never made. The diagnosis was suspected because of the clinical manifestations, laboratory tests, imaging and endoscopic findings. Hyperemic and edematous mucosa of the small intestine corresponded to scattered white spots with dilated intestinal lymphatics and whitish villi in the histological specimen of the biopsied jejunal mucosa. Although numerous therapeutic strategies are available, only octreotide therapy proved to be an effective means of therapeutic resolution in this patient. Although the patient had a partial remission following the use of a slow release formula of octreotide, his prognosis, clinical course, and future treatment challenges are yet to be determined.
Url:
DOI: 10.3748/wjg.v21.i23.7320
PubMed: 26109821
PubMed Central: 4476896
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<front><journal-meta><journal-id journal-id-type="nlm-ta">World J Gastroenterol</journal-id><journal-id journal-id-type="iso-abbrev">World J. Gastroenterol</journal-id><journal-id journal-id-type="publisher-id">WJG</journal-id><journal-title-group><journal-title>World Journal of Gastroenterology : WJG</journal-title></journal-title-group><issn pub-type="ppub">1007-9327</issn><issn pub-type="epub">2219-2840</issn><publisher><publisher-name>Baishideng Publishing Group Inc</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">26109821</article-id><article-id pub-id-type="pmc">4476896</article-id><article-id pub-id-type="other">jWJG.v21.i23.pg7320</article-id><article-id pub-id-type="doi">10.3748/wjg.v21.i23.7320</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>How to treat an extensive form of primary intestinal lymphangiectasia?</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Troskot</surname><given-names>Rosana</given-names></name></contrib><contrib contrib-type="author"><name><surname>Jurčić</surname><given-names>Dragan</given-names></name></contrib><contrib contrib-type="author"><name><surname>Bilić</surname><given-names>Ante</given-names></name></contrib><contrib contrib-type="author"><name><surname>Gomerčić Palčić</surname><given-names>Marija</given-names></name></contrib><contrib contrib-type="author"><name><surname>Težak</surname><given-names>Stanko</given-names></name></contrib><contrib contrib-type="author"><name><surname>Brajković</surname><given-names>Ivana</given-names></name></contrib><aff>Rosana Troskot, Dragan Jurčić, Ante Bilić, Faculty of Medicine, J. J. Strossmayer University of Osijek, Division of Gastroenterology and Hepatology, Department of Internal Medicine, Clinical Hospital “Sveti Duh”, 10000 Zagreb, Croatia</aff><aff>Marija Gomerčić Palčić, Division of Gastroenterology and Hepatology, Department of Internal Medicine, “Sestre milosrdnice” University Hospital Center, University of Zagreb School of Medicine, Vinogradska 29, 10000 Zagreb, Croatia</aff><aff>Stanko Težak, Clinical Department of Nuclear Medicine and Radiation Protection, University Hospital Centre Zagreb, 10000 Zagreb, Croatia</aff><aff>Ivana Brajković, Dialysis clinic, Polyclinic “Sveti Duh” II, 10000 Zagreb, Croatia</aff></contrib-group><author-notes><fn><p>Author contributions: Troskot R, Jurčić D, Bilić A, Gomerčić Palčić M and Brajković I collected the data, performed the literature search and wrote the paper; Težak S participated in drafting the outline and revised the paper.</p><p>Correspondence to: Rosana Troskot, MD, PhD, Faculty of Medicine, J. J. Strossmayer University of Osijek, Division of Gastroenterology and Hepatology, Department of Internal Medicine, Clinical Hospital “Sveti Duh”, Sveti Duh 64, 10000 Zagreb, Croatia. <email>rtroskot@gmail.com</email></p><p>Telephone: +385-1-3712226 Fax: +385-1-3712203</p></fn></author-notes><pub-date pub-type="ppub"><day>21</day><month>6</month><year>2015</year></pub-date><pub-date pub-type="epub"><day>21</day><month>6</month><year>2015</year></pub-date><volume>21</volume><issue>23</issue><fpage>7320</fpage><lpage>7325</lpage><history><date date-type="received"><day>10</day><month>1</month><year>2015</year></date><date date-type="rev-recd"><day>27</day><month>2</month><year>2015</year></date><date date-type="accepted"><day>31</day><month>3</month><year>2015</year></date></history><permissions><copyright-statement>©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.</copyright-statement><copyright-year>2015</copyright-year></permissions><abstract><p>We report a case of a 42-year-old man with a rare disorder known as primary intestinal lymphangiectasia, which is characterized by dilated intestinal lymphatics that lead to the development of protein-losing enteropathy. The patient presented with a grand mal seizure caused by malabsorption-derived electrolytes and a protein disorder. Signs of the disease, including chronic diarrhea and peripheral edema, manifested 10 years ago, but a diagnosis was never made. The diagnosis was suspected because of the clinical manifestations, laboratory tests, imaging and endoscopic findings. Hyperemic and edematous mucosa of the small intestine corresponded to scattered white spots with dilated intestinal lymphatics and whitish villi in the histological specimen of the biopsied jejunal mucosa. Although numerous therapeutic strategies are available, only octreotide therapy proved to be an effective means of therapeutic resolution in this patient. Although the patient had a partial remission following the use of a slow release formula of octreotide, his prognosis, clinical course, and future treatment challenges are yet to be determined.</p></abstract><kwd-group><kwd>Primary intestinal lymphangiectasia</kwd><kwd>Malabsorption</kwd><kwd>Seizure</kwd><kwd>Treatment</kwd><kwd>Diet</kwd><kwd>Octreotide</kwd></kwd-group></article-meta></front></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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