Octreotide in Intestinal Lymphangiectasia: Lack of a Clinical Response and Failure to Alter Lymphatic Function in a Guinea Pig Model

Makhija, Sapna; Weid, P-Y vod der; Meddings, Jon; Sj, Urbanski; Beck, PL

doi:10.1155/2004/176568

Cited by 8 publications

(10 citation statements)

References 30 publications

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“…Octeotride binds to somatostatin receptors in the intestinal lymphatics and decreases bile secretion into and chyle out of the intestine. Tested in vitro , Octreotide did not affect lymphatic function when applied to the mesenteric lymphatic vessel preparation 86 …”

Section: Syndromic Lymphatic Malformationsmentioning

confidence: 99%

Congenital Lymphatic Malformations

Blei

2008

Annals of the New York Academy of Sciences

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"Vascular anomalies" represents a spectrum of vascular lesions, of unclear etiology and often with unpredictable behavior. Patients with vascular anomalies represent a unique population, in that they have focal aberrations of vascular development (in vascular malformations) or vascular proliferation (in hemangiomas). The etiology of these disorders is unclear, and likely represents a multifactorial process. Vascular anomalies are an attractive model for the study of human disorders of vasculogenesis (development of the vasculature) and angiogenesis (new vessel growth from existing vessels).

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Section: Syndromic Lymphatic Malformationsmentioning

confidence: 99%

Congenital Lymphatic Malformations

Blei

2008

Annals of the New York Academy of Sciences

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“…Ten articles described IL in animals (nine articles were for a diagnosis of IL in dogs and one in a horse. One paper was a pharmaceutical experiment on a guinea pig [ 12 ]). The remaining 205 articles described IL in humans.…”

Section: Discussionmentioning

confidence: 99%

Intestinal Lymphangiectasia: Insights on Management and Literature Review

et al. 2016

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Patient: Male, 24Final Diagnosis: Intestinal lymphangiectasiaSymptoms: Frequent episodes of diarrhea • recurrent infections • swelling in the lower limbsMedication: Octreotide • MCT oilsClinical Procedure: Endoscopic exam • Doppler ultrasound study • abdominal CT scanSpecialty: Gastroenterology and HepatologyObjective:Rare diseaseBackground: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.Case Report: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.Conclusions: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.

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“…Intestinal lymphangiectasia is subdivided into primary intestinal lymphangiectasia (PIL) and secondary intestinal lymphangiectasia (Makhija et al 2004). PIL, also known as Waldmann's disease, is a rare disease in humans characterized by dilated lymphatics in the duodenum, jejunum, or mesentery (Buohnik et al 2000;Koo et al 2005).…”

Section: Discussionmentioning

confidence: 99%

Intestinal Lymphangiectasis and Lipidosis in Rats Following Subchronic Exposure to Indole-3-Carbinol via Oral Gavage

et al. 2012

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To investigate the toxicity and carcinogenic potential of indole-3-carbinol (I3C), the National Toxicology Program has conducted 13-week subchronic studies in Fisher 344 rats and B6C3F1 mice, and chronic 2-year bioassays in Sprague-Dawley rats and B6C3F1 mice. While the chronic study resultsare not yet available, subchronic study results and short-term special evaluations of interim sacrifices in the 2-year rat bioassay are presented. F344 rats were orally gavaged 300 mg I3C/kg body weight 5 days a week for 13 weeks. Rats treated with 150 mg/kg demonstrated a dose-related dilation of lymphatics (lymphangiectasis) of the duodenum, jejunum, and mesenteric lymph nodes. Material within dilated lacteals stained positively for Oil Red O and Sudan Black, consistent with lipid. Electron microscopic evaluation confirmed extracellular lipid accumulation within the villar lamina propria, lacteals, and within villar macrophages. Analyses of hepatic and pulmonary CYP1A enzymes demonstrated dose-dependent I3C induction of CYP1A1 and 1A2. B6C3F1 mice orally gavaged 250 mg I3C/kg body weight did not demonstrate histopathological changes; however, hepatic CYP induction was similar to that in rats. The histopathologic changes of intestinal lymphangiectasis and lipidosis in this study share similarities with intestinal lymphangiectasia as observed in humans and dogs. However, the resultant clinical spectrum of protein-losing enteropathy was not present.

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Octreotide in Intestinal Lymphangiectasia: Lack of a Clinical Response and Failure to Alter Lymphatic Function in a Guinea Pig Model

Cited by 8 publications

References 30 publications

Congenital Lymphatic Malformations

Congenital Lymphatic Malformations

Intestinal Lymphangiectasia: Insights on Management and Literature Review

Intestinal Lymphangiectasis and Lipidosis in Rats Following Subchronic Exposure to Indole-3-Carbinol via Oral Gavage

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