MCT 1) 2) 3) 4) 1) 1)2) 3) 4) 1) 1) 1) 1) 1) 5 (albumin Alb ) 2.0g/dl 3 Alb 1.3g/dl 99m (technetium 99m human serum albumin 99m Tc

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1 MCT 1) 2) 3) 4) 1) 1)2) 3) 4) 1) 1) 1) 1) 1) 5 (albumin Alb ) 2.0g/dl 3 Alb 1.3g/dl 99m (technetium 99m human serum albumin 99m TcHSA) (middle chain triglyceride MCT). Key words middle chain triglyceride primary intestinal lymphangiectasia 緒言症例報告. 1). 2) (primary intestinal lymphangiectasia PIL),. 1) 5 (middle chain triglyceride MCT) (albumin Alb) 2.0g/dl / 26 / SpO (room) 65.6cm( 0.3SD) 7.2kg( 0.6SD) Abbreviations: Alb, albumin; 1AT, 1antitrypsin; MCT, middle chain triglyceride; PIL, primary intestinal lymphangiectasia; 99m TcHSA, 99m Tchuman serum albumin

2 51 ( 1) 入院後経過 :( 図 1) 1 60ml 3 Alb1.3g/dl Alb, 1 (antitrypsin AT) 99.7 ( 10mg/ dl ) 3) 99m (techmetium99m human serum albumin 99m TcHSA) 370MBq ( 2) (middle chain triglyceride MCT). 4) ( 2). ( 3) 24 MCT Alb 3.7g/dl MCT. Table 1. Lab Test at the hospitalization CBC Biochemistry Immune Urine WBC Neutro Lymph Mono Eosino Baso Hb Plt 18x x10 4 /µl g/dl /µl Na K CI Alb TBil AST ALT LOH ALP ygtp BS BUN Cr CRP TChol TSH FT3 FT4 Zn Serum Iron Cu Folic Acid Vitamin B12 125Vitamin D3 Vitamin E Vitamin A µu/m ng/dl ng/ml µmol/i TF RBP PreAlb IgG IgA IgM TotallgE Milk ClassO <0.10 U/ml mph 6.0 SG Protein CBC, complete blood count: WBC, white blood cell count; Neutro, neutrophils; Lymph, Iymphocytes; Mono, monocytes; Eosino, eosinophils; Baso, basophils; Hb, hemoglobin; Plt, platelet count; TBil, total bilirubin; AST, aspartate aminotransferase; ALT, alanine aminotranferase; LOH, lactate dehydrogenase; ALp, alkaline phosphatase; ygtp, yglutamyl tanspeptidase; BS, blood sugar; BUN, blood urea nitrogen; Cr, creatinine: CRP, Creative protein; TChol, total cholesterol; TSH, thyroid stimulating hormone; FT3, Free Triiodothyronine; FT4, free thyroxine; TF, Transferrin; RBP, retinolbinding protein; SG, specific gravity

52 Fig.1. Clinical course after hospitalization. is albumin and is protein. Three days after his hospitalization, he got edema and his albumin dropped 1.3g/dl and albumin was supplied.

3 52 Fig.1. Clinical course after hospitalization. is albumin and is protein. Three days after his hospitalization, he got edema and his albumin dropped 1.3g/dl and albumin was supplied. MCT milk was provided since day10 and scintigraphy using 99m Tclabeled human serum albumin was held on day11. Fig.2. 99m TcHSA scintigraphy 11 days after hospitalization. A is 6 hours after taking 99m TcHSA and B is 24 hours after taking 99m TcHSA. Table 2. Lab Tests for investigation Stool Test Rapid Test Bacteria/Virus ph Steatorrhea Fecal Occult Blood Eosinophil α1at(spot stool) < Flu Rotavirus/ Adenovirus /RS virus Blood Culture Stool Culture Ova and Parasite Examination CMV GeniQCMV CD4/CD8 H.pylori IgG<2.0 IgM0.3 <2x20 2 copy/ml 5.3 Negative including urine, stool, and bacterial examinations

4 53 Fig.3. Endoscopy findings. It is provided from National Center for Child Health and Development, Gastroenterology. A is the picture of the duodenal bulb and B is 10 low power field and 40 strong power field. There are some lymphangiectasia in the lamina propria of the duodenal bulb. 考察 PIL PIL MCT 3), PIL 63. 1)5) 10 MCT MCT 22 PIL 3) IgG 200 PIL 6 Munck A 5) 10 PIL ),. 7)8) (quality of life QOL) PIL Alb MCT MCT 9) 10). 614 ( )

5 54 文献 1 ) Xinias I Mavroudi A Sapountzi E Thomaidou A Fotoulaki M Kalambakas A Karypidou E Kollios K Pardalos G Imvrious G Primary intertinal lymphangiectasia: is it always bad? Two cases with different outcome Case Rep Gastroenterol 7: ) 1 Progress of Digestive Endoscopy 59: ) 1 64: ) 40: ) Munck A Sosa Valencia G Faure C Longterm followup of primary intestinal lymphangiectasia in the child Six case reports Arch Pediatr 9: ) Wen J, Tang Q, Wu J, Cai W. Primary intertinal lymphangiectasia: four case reports and a review of the literature. Dig Dis Sci 55: ) Kuroiwa G, Takayama T, Sato Y, Takahashi Y, Fujita T, Nobuoka A, Kukitsu T, Kato J, Sakamaki S, Niitsu Y. Primary intestinal lymphangiectasia successfully treated with octreotide. J Gastroenterol 36: ) 1 58: ) 1 94: ) 1 118: ProteinLosing Gastro Enteropathy Successfully Treated by MCT Milk: a Case Report of an Infant Miho Sodeno, National Center for Global Health and Medicine, Pediatrics. Juzen Med. Soc., 125, (2016) Key words Proteinlosing gastro enteropathy, Primary Intestinal Lymphangiectasia, Hypoalbuminemia, MCT (mediumchain triglyceride) milk Abstract We report a case in a previously healthy 5 months boy of proteinlosing gastro enteropathy, presenting with poor feeding and hypoalbuminemia. At the hospitalization, although his albumin was 2.0g/dl, he had no proteinuria or cardiac dysfunction. Three days after his hospitalization, he got edema and his albumin dropped 1.3g/dl. Scintigraphy using 99m Tclabeled human serum albumin showed the leakage of albumin from the digestive tract and he was diagnosed as Proteinlosing gastro enteropathy. Primary Intestinal Lymphangiectasia was diagnosed, since inflammatory bowel diseases and secondary obstruction of lymph ducts were from the results of blood tests, endoscopy and pathology. The present case was successfully treated by MCT milk. Endoscopy is useful not only for differential diagnosis of proteinlosing gastro enteropathy but also for the improvement of the compliance of the patient for appropriate diet therapy owing to the final diagnosis. In case of children, an early diagnosis and an appropriate treatment is important in order not to cause the delay of growth.

日本呼吸器学会雑誌第48巻第6号

日本呼吸器学会雑誌第48巻第6号 Fig.1 Cutaneousfindingsandpathologicalfindingsofatransbronchiallungbiopsy(TBLB)andlichenifiedeczemaoftherightforearm.Thepatienthadlichenifiedeczemaonhisextremitiesandbodytrunk (1a:rightforearm,1b:leftthigh).Therewasinfiltrationoftheinflammatorylymphocytes,edemaandabrasioninthetype2alveolarepithelium