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[PMID]: 26170774
[Au] Autor:Wu TT; Hoff DS
[Ad] Address:Pharmacy Department, Children's Hospitals and Clinics of Minnesota, Minneapolis, Minnesota.
[Ti] Title:Fish Oil Lipid Emulsion-Associated Sea-Blue Histiocyte Syndrome in a Pediatric Patient.
[So] Source:J Pediatr Pharmacol Ther;20(3):217-21, 2015 May-Jun.
[Is] ISSN:1551-6776
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:A syndrome of hepatosplenomegaly, thrombocytopenia, and anemia and the presence of sea-blue histiocytes in bone marrow has been associated with parenteral soybean oil administration in patients receiving long-term total parenteral nutrition (TPN). A case is described here where this syndrome was observed in a pediatric patient who received long-term parenteral fish oil nutrition.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1507
[Cu] Class update date: 170220
[Lr] Last revision date:170220
[Da] Date of entry for processing:150715
[St] Status:PubMed-not-MEDLINE
[do] DOI:10.5863/1551-6776-20.3.217

  2 / 131 MEDLINE  
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[PMID]: 25413372
[Au] Autor:Bermejo N; Prieto J; Arcos MJ
[Ad] Address:Department of Hematology, Hospital San Pedro de Alcántara, Cáceres, Spain.
[Ti] Title:Sea-blue histiocytosis in bone marrow of a patient with chronic thrombocytopenia.
[So] Source:Acta Haematol;133(3):277-8, 2015.
[Is] ISSN:1421-9662
[Cp] Country of publication:Switzerland
[La] Language:eng
[Mh] MeSH terms primary: Bone Marrow/pathology
Sea-Blue Histiocyte Syndrome/pathology
Thrombocytopenia/pathology
[Mh] MeSH terms secundary: Chronic Disease
Humans
Middle Aged
Sea-Blue Histiocyte Syndrome/complications
Thrombocytopenia/complications
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Em] Entry month:1506
[Cu] Class update date: 150415
[Lr] Last revision date:150415
[Js] Journal subset:IM
[Da] Date of entry for processing:141122
[St] Status:MEDLINE
[do] DOI:10.1159/000366189

  3 / 131 MEDLINE  
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[PMID]: 24787480
[Au] Autor:Michot JM; Gubavu C; Fourn E; Maigne G; Teicher E; Angoulvant A; Blanche S; Lortholary O; Coilly A; Duclos-Vallée JC; Sebagh M; Guettier C; Aumont C; Delfraissy JF; Lambotte O
[Ad] Address:Assistance Publique - Hôpitaux de Paris, Service de Médecine Interne et Immunologie Clinique, Hôpital Universitaire Bicêtre, 94275 Le Kremlin-Bicêtre Cedex, France; Université Paris Sud XI, 63 rue Gabriel Péri, 94276 Le Kremlin-Bicêtre Cedex, France. Electronic address: jean-marie.michot@bct.aphp.fr
[Ti] Title:Very prolonged liposomal amphotericin B use leading to a lysosomal storage disease.
[So] Source:Int J Antimicrob Agents;43(6):566-9, 2014 Jun.
[Is] ISSN:1872-7913
[Cp] Country of publication:Netherlands
[La] Language:eng
[Ab] Abstract:Amphotericin B is a powerful polyene antifungal drug used for treating systemic fungal infections and is usually administered for a short period. Side effects after prolonged use are unknown in humans. Here we report the case of a 28-year-old man suffering from chronic granulomatous disease (CGD), treated for invasive cerebral aspergillosis with liposomal amphotericin B (L-AmB) for a very long time (8 consecutive years). We describe the efficacy and safety of this treatment in the long term. Aspergillosis was kept under control as long as L-AmB therapy was maintained, but relapsed when the dose was reduced. No overt renal toxicity was noted. The patient gradually developed hepatosplenomegaly and pancytopenia. Abnormalities of bone marrow were similar to the sea-blue histiocyte syndrome. Liver biopsy showed images of nodular regenerative hyperplasia related to CGD as well as a histiocytic storage disease. We discuss the very prolonged use of L-AmB leading to the development of a lysosomal storage disease.
[Mh] MeSH terms primary: Amphotericin B/adverse effects
Amphotericin B/therapeutic use
Antifungal Agents/adverse effects
Antifungal Agents/therapeutic use
Lysosomal Storage Diseases/chemically induced
[Mh] MeSH terms secundary: Adult
Biopsy
Granulomatous Disease, Chronic/complications
Histocytochemistry
Humans
Liver/pathology
Male
Neuroaspergillosis/drug therapy
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:0 (Antifungal Agents); 0 (liposomal amphotericin B); 7XU7A7DROE (Amphotericin B)
[Em] Entry month:1501
[Cu] Class update date: 140609
[Lr] Last revision date:140609
[Js] Journal subset:IM
[Da] Date of entry for processing:140503
[St] Status:MEDLINE

  4 / 131 MEDLINE  
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[PMID]: 22779941
[Au] Autor:Günay E; Firat Güven S; Aktas Z; Sipit T; Agaçkiran Y; Ertürk H
[Ad] Address:Clinic of Chest Diseases, Ataturk Chest Diseases and Chest Surgery Training and Research Hospital, Ankara, Turkey. ersingunay@gmail.com.
[Ti] Title:Pulmonary involvement in sea-blue histiocytosis.
[So] Source:Tuberk Toraks;60(2):176-9, 2012.
[Is] ISSN:0494-1373
[Cp] Country of publication:Turkey
[La] Language:eng
[Ab] Abstract:Sea-blue histiocytosis is one of the six types of Niemann-Pick disease. It is characterized by childhood onset of hepatosplenomegaly, lack of neurological involvement and diminished sphingomyelinase activity. Pulmonary system is rarely involved sea-blue histiocytosis. In this paper, we present a 39-years-old male who had previously diagnosed as sea-blue histiocytosis at the age of 15. He was admitted to our clinic due to productive cough, hemoptysis, fever and weight loss. His symptoms did not resolve with the antibiotic treatment and further investigations revealed pulmonary involvement of sea-blue histiocytosis. After diagnostic bronchoalveolar lavage, his symptoms were improved, interestingly. This rare entity was discussed with literature survey.
[Mh] MeSH terms primary: Bronchoalveolar Lavage
Lung Diseases/etiology
Sea-Blue Histiocyte Syndrome/complications
[Mh] MeSH terms secundary: Adult
Humans
Lung Diseases/therapy
Male
Treatment Outcome
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Em] Entry month:1209
[Cu] Class update date: 130528
[Lr] Last revision date:130528
[Js] Journal subset:IM
[Da] Date of entry for processing:120712
[St] Status:MEDLINE

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[PMID]: 20630517
[Au] Autor:Safatle-Ribeiro AV; Baba ER; Iriya K; Fylyk SN; Pennacchi C; Couto Junior DS; de Moura EG; Sakai P
[Ad] Address:Department of Gastroenterology, University of São Paulo School of Medicine, São Paulo, Brazil.
[Ti] Title:Double-balloon endoscopy reveals sea-blue histiocytosis affecting the small bowel (with video).
[So] Source:Gastrointest Endosc;72(6):1266; discussion 1266-7, 2010 Dec.
[Is] ISSN:1097-6779
[Cp] Country of publication:United States
[La] Language:eng
[Mh] MeSH terms primary: Double-Balloon Enteroscopy
Duodenal Diseases/diagnosis
Intestinal Diseases/diagnosis
Intestine, Small
Sea-Blue Histiocyte Syndrome/diagnosis
[Mh] MeSH terms secundary: Biopsy
Diagnosis, Differential
Duodenal Diseases/pathology
Duodenum/pathology
Female
Humans
Intestinal Diseases/pathology
Intestine, Small/pathology
Young Adult
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE; VIDEO-AUDIO MEDIA
[Em] Entry month:1104
[Cu] Class update date: 101129
[Lr] Last revision date:101129
[Js] Journal subset:IM
[Da] Date of entry for processing:100716
[St] Status:MEDLINE
[do] DOI:10.1016/j.gie.2010.05.024

  6 / 131 MEDLINE  
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[PMID]: 19864213
[Au] Autor:Sharma P; Kar R; Dutta S; Pati HP; Saxena R
[Ad] Address:Haematology Department, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, India. prashant.sh@gmail.com
[Ti] Title:Niemann-Pick disease, type B with TRAP-positive storage cells and secondary sea blue histiocytosis.
[So] Source:Eur J Histochem;53(3):183-6, 2009 Sep 23.
[Is] ISSN:1121-760X
[Cp] Country of publication:Italy
[La] Language:eng
[Ab] Abstract:We present 2 cases of Niemann Pick disease, type B with secondary sea-blue histiocytosis. Strikingly, in both cases the Pick cells were positive for tartrate resistant acid phosphatase, a finding hitherto described only in Gaucher cells. This report highlights the importance of this finding as a potential cytochemical diagnostic pitfall in the diagnosis of Niemann Pick disease.
[Mh] MeSH terms primary: Acid Phosphatase/analysis
Isoenzymes/analysis
Niemann-Pick Disease, Type B/complications
Sea-Blue Histiocyte Syndrome/complications
[Mh] MeSH terms secundary: Adolescent
Female
Humans
Niemann-Pick Disease, Type B/pathology
Sea-Blue Histiocyte Syndrome/pathology
Tartrate-Resistant Acid Phosphatase
[Pt] Publication type:CASE REPORTS; LETTER
[Nm] Name of substance:0 (Isoenzymes); EC 3.1.3.2 (Acid Phosphatase); EC 3.1.3.2 (Tartrate-Resistant Acid Phosphatase)
[Em] Entry month:0911
[Cu] Class update date: 161125
[Lr] Last revision date:161125
[Js] Journal subset:IM
[Da] Date of entry for processing:091030
[St] Status:MEDLINE

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SciELO Spain full text

[PMID]: 19721912
[Au] Autor:Egaña N; Parón L; Cuerda C; Bretón I; Camblor M; Velasco C; García-Peris P
[Ad] Address:Unidad de Nutrición, Hospital General Universitario Gregorio Marañón, Madrid, España.
[Ti] Title:Síndrome del histiocito azul marino en relación a nutrición parenteral domiciliaria. [Sea-blue histiocyte syndrome associated with home parenteral nutrition].
[So] Source:Nutr Hosp;24(3):361-3, 2009 May-Jun.
[Is] ISSN:0212-1611
[Cp] Country of publication:Spain
[La] Language:spa
[Ab] Abstract:A case of a 55 years-old male with long-term Crohn's disease without response to medical treatment and many intestinal fistula is presented. After the last bowel resection, home parenteral nutrition was started. He presented chronic hepatopathy and pancytopaenia. After 9 months of home parenteral nutrition hepatic function and pancytopaenia began to deteriorate. Bone marrow examination revealed an infiltrate of sea-blue histiocytes. He made unsatisfactory progress and died due to a multiorganic failure.
[Mh] MeSH terms primary: Parenteral Nutrition, Home/adverse effects
Sea-Blue Histiocyte Syndrome/etiology
[Mh] MeSH terms secundary: Humans
Male
Middle Aged
[Pt] Publication type:CASE REPORTS; ENGLISH ABSTRACT; JOURNAL ARTICLE
[Em] Entry month:0909
[Cu] Class update date: 130702
[Lr] Last revision date:130702
[Js] Journal subset:IM
[Da] Date of entry for processing:090902
[St] Status:MEDLINE

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[PMID]: 19592052
[Au] Autor:Naghashpour M; Cualing H
[Ad] Address:Division of Hematopathology and Laboratory Medicine, Department of Oncologic Science, H. Lee Moffitt Cancer Center, University of South Florida, USA. mojdeh.naghashpour@moffitt.org
[Ti] Title:Splenomegaly with sea-blue histiocytosis, dyslipidemia, and nephropathy in a patient with lecithin-cholesterol acyltransferase deficiency: a clinicopathologic correlation.
[So] Source:Metabolism;58(10):1459-64, 2009 Oct.
[Is] ISSN:1532-8600
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:A 31-year-old man with no significant medical history presented with a 5-day history of progressive left upper quadrant abdominal pain. Physical examination revealed a tender guarded abdomen, no icterus, and bilateral corneal "arcus senilis"-like changes. Laboratory workup showed a mild normocytic, normochromic anemia; and target cells were seen in the peripheral blood smear. Serum was turbid; and the lipid profile showed elevated total cholesterol, low high-density lipoprotein cholesterol, and elevated triglycerides. Urinalysis revealed nephrotic range proteinuria with microhematuria. An abdominal computed tomographic scan demonstrated a homogeneously enlarged spleen. The patient was discharged after symptomatic treatment to be followed as an ambulatory patient. Several days later, he returned with severe left upper quadrant pain and was admitted to the surgical service for further evaluation. A splenectomy was performed for a suspected splenic lymphoma. Upon gross examination, spleen was moderately enlarged, weighing 780 g. Sectioning revealed a beefy red cut surface without gross lesions. Wright-Giemsa-stained touch imprints showed many sea-blue histiocytes. A renal biopsy was also performed, demonstrating focal segmental glomerular sclerosis and mesangial expansion with extramembranous and intramembranous deposition of lipids. In the absence of hematologic malignancy and in light of the abnormal lipid profile, a disorder of lipid metabolism was suspected. Histologic and ultrastructural findings in the kidney and spleen raised the likelihood of lecithin-cholesterol acyltransferase (LCAT) deficiency, which was confirmed by the markedly decreased serum LCAT activity and serum LCAT mass. We describe a case with the triad of splenomegaly with sea-blue histiocytes, nephropathy, and dyslipidemia in a patient with LCAT deficiency.
[Mh] MeSH terms primary: Dyslipidemias/blood
Kidney Diseases/pathology
Lecithin Cholesterol Acyltransferase Deficiency/pathology
Sea-Blue Histiocyte Syndrome/pathology
Splenomegaly/pathology
[Mh] MeSH terms secundary: Adult
Blood Cell Count
Dyslipidemias/complications
Humans
Kidney/pathology
Kidney Diseases/complications
Lecithin Cholesterol Acyltransferase Deficiency/blood
Lecithin Cholesterol Acyltransferase Deficiency/complications
Liver/pathology
Male
Microscopy, Electron, Transmission
Phosphatidylcholine-Sterol O-Acyltransferase/blood
Sea-Blue Histiocyte Syndrome/complications
Spleen/pathology
Splenomegaly/complications
Tomography, X-Ray Computed
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:EC 2.3.1.43 (Phosphatidylcholine-Sterol O-Acyltransferase)
[Em] Entry month:0910
[Cu] Class update date: 161125
[Lr] Last revision date:161125
[Js] Journal subset:IM
[Da] Date of entry for processing:090714
[St] Status:MEDLINE
[do] DOI:10.1016/j.metabol.2009.04.033

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[PMID]: 19411774
[Au] Autor:Cho YU; Chae JD; Lee WM; Woo JJ; Lee HB; Gong SJ; Park CJ; Kim GH; Yoo HW
[Ad] Address:Department of Laboratory Medicine, Eulji University School of Medicine, Eulji General Hospital, Nowon-gu, Seoul, Korea.
[Ti] Title:[A case of a Korean adult affected by type B Niemann-Pick disease: secondary sea-blue histiocytosis and molecular characterization].
[So] Source:Korean J Lab Med;29(2):97-103, 2009 Apr.
[Is] ISSN:1598-6535
[Cp] Country of publication:Korea (South)
[La] Language:kor
[Ab] Abstract:Niemann-Pick disease (NPD) is an inherited metabolic disorder caused by a deficiency of the enzyme acid sphingomyelinase coded by SMPD1 gene. In contrast with type A NPD, a severe neurodegenerative disease of infancy, type B NPD patients have little or no neurodegeneration, and frequently survive into adulthood. Although over 100 mutations have been found within the SMPD1 gene causing NPD, there was only one report about SMPD1 mutation status of a Korean NPD patient. We report a case of a 32-yr-old female, who presented with thrombocytopenia without any neurologic involvement. Hepatosplenomegaly was detected by both physical examination and imaging studies, and a thoracic radiograph examination showed a pattern of interstitial lung disease. Biochemical tests revealed increased liver enzymes, cholesterol, triglyceride, and LDL-cholesterol, and decreased HDL-cholesterol. Sea-blue or foamy vacuolated histiocytes occurred in bone marrow and liver. Sequencing analysis of SMPD1 using genomic DNA from peripheral leukocytes identified a compound heterozygote of two mutations at exon 2: p.E246K and p.A357V. The former is a known mutation in an Italian patient, and the latter has not been reported yet. She has received oral rosuvastatin to treat hyperlipidemia at a dose of 10 mg per day for 4 months. This is the second report in which the mutation of SMPD1 gene was detected in a Korean NPD patient. The active genetic analysis of SMPD1 gene in patients with typical findings of type B NPD would enable us to facilitate diagnosis as well as to accumulate data on molecular characteristics of Korean NPD patients.
[Mh] MeSH terms primary: Niemann-Pick Disease, Type B/diagnosis
[Mh] MeSH terms secundary: Adult
Base Sequence
Bone Marrow Cells/pathology
Female
Humans
Korea
Liver/pathology
Niemann-Pick Disease, Type B/genetics
Niemann-Pick Disease, Type B/radiotherapy
Pregnancy
Sea-Blue Histiocyte Syndrome/diagnosis
Sea-Blue Histiocyte Syndrome/pathology
Sequence Analysis, DNA
Sphingomyelin Phosphodiesterase/genetics
Tomography, X-Ray Computed
[Pt] Publication type:CASE REPORTS; ENGLISH ABSTRACT; JOURNAL ARTICLE
[Nm] Name of substance:EC 3.1.4.12 (Sphingomyelin Phosphodiesterase)
[Em] Entry month:0908
[Cu] Class update date: 140530
[Lr] Last revision date:140530
[Js] Journal subset:IM
[Da] Date of entry for processing:090505
[St] Status:MEDLINE
[do] DOI:10.3343/kjlm.2009.29.2.97

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[PMID]: 17510534
[Au] Autor:Suzuki O; Abe M
[Ad] Address:First Department of Pathology, School of Medicine, Fukushima Medical University, Fukushima, Japan. osuzuki@fmu.ac.jp
[Ti] Title:Secondary sea-blue histiocytosis derived from Niemann-Pick disease.
[So] Source:J Clin Exp Hematop;47(1):19-21, 2007 Apr.
[Is] ISSN:1346-4280
[Cp] Country of publication:Japan
[La] Language:eng
[Ab] Abstract:Sea-blue histiocytosis is a rare disorder seen in patients with lipid metabolic or ceroid storage diseases. Sea-blue histiocytes are ceroid-laden macrophages detectable by May-Giemsa staining. We report a case of a 28-year-old woman diagnosed with Niemann-Pick disease at 2 or 3 years of age. To confirm this diagnosis, we examined her bone marrow, which revealed scattered foci containing aggregates of foamy macrophages. May-Giemsa staining identified blue-staining foamy macrophages, referred to as sea-blue histiocytes. In summary, we report the detection of sea-blue histiocytosis in an adult with Niemann-Pick disease.
[Mh] MeSH terms primary: Bone Marrow Cells/pathology
Macrophages/pathology
Niemann-Pick Diseases/complications
Sea-Blue Histiocyte Syndrome/etiology
[Mh] MeSH terms secundary: Adult
Bone Marrow Examination
Female
Humans
Immunohistochemistry
Leukocytes/enzymology
Niemann-Pick Diseases/diagnosis
Sea-Blue Histiocyte Syndrome/diagnosis
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Em] Entry month:0707
[Cu] Class update date: 070518
[Lr] Last revision date:070518
[Js] Journal subset:IM
[Da] Date of entry for processing:070519
[St] Status:MEDLINE


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