Intestinal Lymphangiectasia: Insights on Management and Literature Review
Identifieur interne : 000F43 ( Main/Exploration ); précédent : 000F42; suivant : 000F44Intestinal Lymphangiectasia: Insights on Management and Literature Review
Auteurs : Mohamad J. Alshikho [États-Unis] ; Joud M. Talas ; Salem I. Noureldine [États-Unis] ; Saf Zazou ; Aladdin Addas ; Haitham Kurabi ; Mahmoud NasserSource :
- The American Journal of Case Reports [ 1941-5923 ] ; 2016.
Abstract
Intestinal lymphangiectasia (IL) is a rare disease characterized by a dilatation of the intestinal lymphatics and loss of lymph fluid into the gastrointestinal tract leading to hypoproteinemia, edema, lymphocytopenia, hypogammaglobinemia, and immunological abnormalities. Iron, calcium, and other serum components (e.g., lipids, fat soluble vitamins) may also be depleted. A literature search revealed more than 200 reported cases of IL. Herein, we report our observations of a patient diagnosed with IL; we also present our conclusion for our review of the published literature.
A 24-year-old male was admitted to Aleppo University Hospital with the complaints of abdominal pain, headache, arthralgia, fever, and rigors. His past medical history was remarkable for frequent episodes of diarrhea, recurrent infections, and swelling in the lower limbs. In addition, he had been hospitalized several times in non-academic hospitals due to edema in his legs, cellulitis, and recurrent infections. In the emergency department, a physical examination revealed a patient in distress. He was weak, dehydrated, pale, and had a high-grade fever. His lower extremities were edematous, swollen, and extremely tender to touch. The overlying skin was erythematous and warm. Moreover, the patient was tachycardic, tacypneic, and moderately hypotensive. The patient was resuscitated with IV fluids, and Tylenol was administered to bring the temperature down. Blood tests showed anemia and high levels of inflammatory markers. The patient’s white blood cell count was elevated with an obvious left shift. However, subsequent investigations showed that the patient had IL. Suitable diet modification plans were applied as a long-term management plan.
IL is a rare disease of challenging nature due to its systematic effects and lack of comprehensive studies that can evaluate the effectiveness of specific treatments in a large cohort of patients. Medium-chain triglyceride (MCT) oils and diet modification strategies are effective in reducing the loss of body proteins and in maintaining near-normal blood levels of immunoglobulins. However, octreotide and MCT oils had no proven role in reducing lymphedema in our patient.
Url:
DOI: 10.12659/AJCR.899636
PubMed: 27440277
PubMed Central: 4957630
Affiliations:
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Alshikho</name><affiliation wicri:level="4"><nlm:aff id="af1-amjcaserep-17-512">Department of Neurology, Massachusetts General Hospital, Harvard University, Boston, MA, U.S.A.</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Massachusetts General Hospital, Harvard University, Boston, MA</wicri:regionArea><orgName type="university">Université Harvard</orgName><placeName><settlement type="city">Cambridge (Massachusetts)</settlement><region type="state">Massachusetts</region></placeName></affiliation></author><author><name sortKey="Talas, Joud M" sort="Talas, Joud M" uniqKey="Talas J" first="Joud M." last="Talas">Joud M. Talas</name><affiliation><nlm:aff id="af2-amjcaserep-17-512">Department of Dermatology, Aleppo University, Aleppo, Syria</nlm:aff><wicri:noCountry code="subfield">Syria</wicri:noCountry></affiliation></author><author><name sortKey="Noureldine, Salem I" sort="Noureldine, Salem I" uniqKey="Noureldine S" first="Salem I." last="Noureldine">Salem I. Noureldine</name><affiliation wicri:level="1"><nlm:aff id="af3-amjcaserep-17-512">Department of Otolaryngology – Head and Neck Surgery, Johns Hopkins University, Baltimore, MD, U.S.A.</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Otolaryngology – Head and Neck Surgery, Johns Hopkins University, Baltimore, MD</wicri:regionArea><wicri:noRegion>MD</wicri:noRegion></affiliation></author><author><name sortKey="Zazou, Saf" sort="Zazou, Saf" uniqKey="Zazou S" first="Saf" last="Zazou">Saf Zazou</name><affiliation><nlm:aff id="af4-amjcaserep-17-512">Department of General Internal Medicine, Aleppo University, Aleppo, Syria</nlm:aff><wicri:noCountry code="subfield">Syria</wicri:noCountry></affiliation></author><author><name sortKey="Addas, Aladdin" sort="Addas, Aladdin" uniqKey="Addas A" first="Aladdin" last="Addas">Aladdin Addas</name><affiliation><nlm:aff id="af4-amjcaserep-17-512">Department of General Internal Medicine, Aleppo University, Aleppo, Syria</nlm:aff><wicri:noCountry code="subfield">Syria</wicri:noCountry></affiliation></author><author><name sortKey="Kurabi, Haitham" sort="Kurabi, Haitham" uniqKey="Kurabi H" first="Haitham" last="Kurabi">Haitham Kurabi</name><affiliation><nlm:aff id="af4-amjcaserep-17-512">Department of General Internal Medicine, Aleppo University, Aleppo, Syria</nlm:aff><wicri:noCountry code="subfield">Syria</wicri:noCountry></affiliation></author><author><name sortKey="Nasser, Mahmoud" sort="Nasser, Mahmoud" uniqKey="Nasser M" first="Mahmoud" last="Nasser">Mahmoud Nasser</name><affiliation><nlm:aff id="af5-amjcaserep-17-512">Department of Gastroenterology, Aleppo University, Aleppo, Syria</nlm:aff><wicri:noCountry code="subfield">Syria</wicri:noCountry></affiliation></author></analytic><series><title level="j">The American Journal of Case Reports</title><idno type="eISSN">1941-5923</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p><bold>Patient: Male, 24</bold></p><p><bold>Final Diagnosis: Intestinal lymphangiectasia</bold></p><p><bold>Symptoms: Frequent episodes of diarrhea • recurrent infections • swelling in the lower limbs</bold></p><p><bold>Medication: Octreotide • MCT oils</bold></p><p><bold>Clinical Procedure: Endoscopic exam • Doppler ultrasound study • abdominal CT scan</bold></p><p><bold>Specialty: Gastroenterology and Hepatology</bold></p><sec><title>Objective:</title><p><bold>Rare disease</bold></p></sec><sec><title>Background:</title><p>Intestinal lymphangiectasia (IL) is a rare disease characterized by a dilatation of the intestinal lymphatics and loss of lymph fluid into the gastrointestinal tract leading to hypoproteinemia, edema, lymphocytopenia, hypogammaglobinemia, and immunological abnormalities. Iron, calcium, and other serum components (e.g., lipids, fat soluble vitamins) may also be depleted. A literature search revealed more than 200 reported cases of IL. Herein, we report our observations of a patient diagnosed with IL; we also present our conclusion for our review of the published literature.</p></sec><sec><title>Case Report:</title><p>A 24-year-old male was admitted to Aleppo University Hospital with the complaints of abdominal pain, headache, arthralgia, fever, and rigors. His past medical history was remarkable for frequent episodes of diarrhea, recurrent infections, and swelling in the lower limbs. In addition, he had been hospitalized several times in non-academic hospitals due to edema in his legs, cellulitis, and recurrent infections. In the emergency department, a physical examination revealed a patient in distress. He was weak, dehydrated, pale, and had a high-grade fever. His lower extremities were edematous, swollen, and extremely tender to touch. The overlying skin was erythematous and warm. Moreover, the patient was tachycardic, tacypneic, and moderately hypotensive. The patient was resuscitated with IV fluids, and Tylenol was administered to bring the temperature down. Blood tests showed anemia and high levels of inflammatory markers. The patient’s white blood cell count was elevated with an obvious left shift. However, subsequent investigations showed that the patient had IL. Suitable diet modification plans were applied as a long-term management plan.</p></sec><sec><title>Conclusions:</title><p>IL is a rare disease of challenging nature due to its systematic effects and lack of comprehensive studies that can evaluate the effectiveness of specific treatments in a large cohort of patients. Medium-chain triglyceride (MCT) oils and diet modification strategies are effective in reducing the loss of body proteins and in maintaining near-normal blood levels of immunoglobulins. However, octreotide and MCT oils had no proven role in reducing lymphedema in our patient.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Vignes, S" uniqKey="Vignes S">S Vignes</name></author><author><name sortKey="Carcelain, G" uniqKey="Carcelain G">G Carcelain</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Vignes, S" uniqKey="Vignes S">S Vignes</name></author><author><name sortKey="Bellanger, J" uniqKey="Bellanger J">J Bellanger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Umar, Sb" uniqKey="Umar S">SB Umar</name></author><author><name sortKey="Dibaise, Jk" uniqKey="Dibaise J">JK DiBaise</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Malek, Np" uniqKey="Malek N">NP Malek</name></author><author><name sortKey="Ocran, K" uniqKey="Ocran K">K Ocran</name></author><author><name sortKey="Tietge, Uj" uniqKey="Tietge 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sortKey="Sato, Y" uniqKey="Sato Y">Y Sato</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Fleisher, Ta" uniqKey="Fleisher T">TA Fleisher</name></author><author><name sortKey="Strober, W" uniqKey="Strober W">W Strober</name></author><author><name sortKey="Muchmore, Av" uniqKey="Muchmore A">AV Muchmore</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Alfano, V" uniqKey="Alfano V">V Alfano</name></author><author><name sortKey="Tritto, G" uniqKey="Tritto G">G Tritto</name></author><author><name sortKey="Alfonsi, L" uniqKey="Alfonsi L">L Alfonsi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Barrett, Ds" uniqKey="Barrett D">DS Barrett</name></author><author><name sortKey="Large, Sr" uniqKey="Large S">SR Large</name></author><author><name sortKey="Rees, Gm" uniqKey="Rees G">GM Rees</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Fadell, Ej" uniqKey="Fadell E">EJ Fadell</name></author><author><name sortKey="Dame, Rw" uniqKey="Dame R">RW Dame</name></author><author><name sortKey="Wolford, Jl" uniqKey="Wolford J">JL Wolford</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Makhija, S" uniqKey="Makhija S">S Makhija</name></author><author><name sortKey="Von Der Weid, Py" uniqKey="Von Der Weid P">PY von der Weid</name></author><author><name sortKey="Meddings, J" uniqKey="Meddings J">J Meddings</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Altit, G" uniqKey="Altit G">G Altit</name></author><author><name sortKey="Patel, H" uniqKey="Patel H">H Patel</name></author><author><name sortKey="Morinville, Vd" uniqKey="Morinville V">VD Morinville</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kuroiwa, G" uniqKey="Kuroiwa G">G Kuroiwa</name></author><author><name sortKey="Takayama, T" uniqKey="Takayama T">T Takayama</name></author><author><name sortKey="Sato, Y" uniqKey="Sato Y">Y Sato</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Filik, L" uniqKey="Filik L">L Filik</name></author><author><name sortKey="Oguz, P" uniqKey="Oguz P">P Oguz</name></author><author><name sortKey="Koksal, A" uniqKey="Koksal A">A Koksal</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Klingenberg, Rd" uniqKey="Klingenberg R">RD Klingenberg</name></author><author><name sortKey="Homann, N" uniqKey="Homann N">N Homann</name></author><author><name sortKey="Ludwig, D" uniqKey="Ludwig D">D Ludwig</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Bac, Dj" uniqKey="Bac D">DJ Bac</name></author><author><name sortKey="Van Hagen, Pm" uniqKey="Van Hagen P">PM Van Hagen</name></author><author><name sortKey="Postema, Pt" uniqKey="Postema P">PT Postema</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Suehiro, K" uniqKey="Suehiro K">K Suehiro</name></author><author><name sortKey="Morikage, N" uniqKey="Morikage N">N Morikage</name></author><author><name sortKey="Murakami, M" uniqKey="Murakami M">M Murakami</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Al Sinani, S" uniqKey="Al Sinani S">S Al Sinani</name></author><author><name sortKey="Rawahi, Ya" uniqKey="Rawahi Y">YA Rawahi</name></author><author><name sortKey="Abdoon, H" uniqKey="Abdoon H">H Abdoon</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Balboa, A" uniqKey="Balboa A">A Balboa</name></author><author><name sortKey="Perello, A" uniqKey="Perello A">A Perello</name></author><author><name sortKey="Mearin, F" uniqKey="Mearin F">F Mearin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ballinger, Ab" uniqKey="Ballinger A">AB 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sortKey="Diuguid, Dl" uniqKey="Diuguid D">DL Diuguid</name></author><author><name sortKey="Whitlock, Rt" uniqKey="Whitlock R">RT Whitlock</name></author><author><name sortKey="Holt, Pr" uniqKey="Holt P">PR Holt</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hargrove, Md" uniqKey="Hargrove M">MD Hargrove</name></author><author><name sortKey="Mathews, Wr" uniqKey="Mathews W">WR Mathews</name></author><author><name sortKey="Mcintyre, Pa" uniqKey="Mcintyre P">PA McIntyre</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Fleisher, Ta" uniqKey="Fleisher T">TA Fleisher</name></author><author><name sortKey="Strober, W" uniqKey="Strober W">W Strober</name></author><author><name sortKey="Muchmore, Av" uniqKey="Muchmore A">AV Muchmore</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Alfano, V" uniqKey="Alfano V">V Alfano</name></author><author><name sortKey="Tritto, G" uniqKey="Tritto G">G Tritto</name></author><author><name sortKey="Alfonsi, L" uniqKey="Alfonsi L">L Alfonsi</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Massachusetts</li></region><settlement><li>Cambridge (Massachusetts)</li></settlement><orgName><li>Université Harvard</li></orgName></list><tree><noCountry><name sortKey="Addas, Aladdin" sort="Addas, Aladdin" uniqKey="Addas A" first="Aladdin" last="Addas">Aladdin Addas</name><name sortKey="Kurabi, Haitham" sort="Kurabi, Haitham" uniqKey="Kurabi H" first="Haitham" last="Kurabi">Haitham Kurabi</name><name sortKey="Nasser, Mahmoud" sort="Nasser, Mahmoud" uniqKey="Nasser M" first="Mahmoud" last="Nasser">Mahmoud Nasser</name><name sortKey="Talas, Joud M" sort="Talas, Joud M" uniqKey="Talas J" first="Joud M." last="Talas">Joud M. Talas</name><name sortKey="Zazou, Saf" sort="Zazou, Saf" uniqKey="Zazou S" first="Saf" last="Zazou">Saf Zazou</name></noCountry><country name="États-Unis"><region name="Massachusetts"><name sortKey="Alshikho, Mohamad J" sort="Alshikho, Mohamad J" uniqKey="Alshikho M" first="Mohamad J." last="Alshikho">Mohamad J. Alshikho</name></region><name sortKey="Noureldine, Salem I" sort="Noureldine, Salem I" uniqKey="Noureldine S" first="Salem I." last="Noureldine">Salem I. Noureldine</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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