RSS-Feed abonnieren
DOI: 10.1055/a-2559-4147
Sea-blue histiocytosis: an unusual small-bowel endoscopic finding
A 67-year-old man, diagnosed with heart failure and atrial fibrillation, was referred for anterograde double-balloon enteroscopy to investigate iron deficiency anemia requiring blood transfusion and duodenal polyposis diagnosed on upper digestive endoscopy. Colonoscopy was normal. The patient also had chronic liver disease, with small esophageal varices, hepatosplenomegaly, and thrombocytopenia. Laboratory examination showed hemoglobin of 8.6 g/dL, alkaline phosphatase 207 IU/L, and gamma-glutamyl transferase 83 IU/L.
Double-balloon enteroscopy revealed numerous sessile, elevated, pedunculated, and multilobulated lesions, some forming bridges into the lumen, distributed throughout the duodenum and the entire portion of the examined proximal and mid jejunum ([Fig. 1], [Video 1]).
Qualität:
Histology of duodenal and jejunal biopsy samples demonstrated subepithelial foamy macrophages with cytoplasmic granularity on hematoxylin and eosin staining ([Fig. 2] a). Due to the accumulation of mucosubstances including glycolipids in the cytoplasm, the histiocytes appeared with lipopigment granules that were stained by periodic acid–Schiff ([Fig. 2] b) and by Ziehl–Neelsen stain ([Fig. 2] c). No bacteria were observed.
Sea-blue histiocytosis has already been described in several clinical conditions such as lipid storage diseases (Niemann–Pick), thalassemia, myelodysplastic and myeloproliferative syndromes, liver diseases, and in patients on parenteral nutrition [1] [2]. Sea-blue histiocytosis is a very rare lysosomal lipid storage disease that can be found in various organs such as bone marrow, liver, spleen, and small intestine [3]. Possible differential diagnoses are Gaucher disease and Whipple disease [2]. Thrombocytopenia, anemia, splenomegaly, and hepatomegaly may be present, and a few cases may progress to liver cirrhosis and portal hypertension [4] [5].
Endoscopy_UCTN_Code_CCL_1AC_2AH
E-Videos is an open access online section of the journal Endoscopy, reporting on interesting cases and new techniques in gastroenterological endoscopy. All papers include a high-quality video and are published with a Creative Commons CC-BY license. Endoscopy E-Videos qualify for HINARI discounts and waivers and eligibility is automatically checked during the submission process. We grant 100% waivers to articles whose corresponding authors are based in Group A countries and 50% waivers to those who are based in Group B countries as classified by Research4Life (see: https://www.research4life.org/access/eligibility/).
This section has its own submission website at https://mc.manuscriptcentral.com/e-videos.
Publikationsverlauf
Artikel online veröffentlicht:
27. März 2025
© 2025. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution License, permitting unrestricted use, distribution, and reproduction so long as the original work is properly cited. (https://creativecommons.org/licenses/by/4.0/).
Georg Thieme Verlag KG
Oswald-Hesse-Straße 50, 70469 Stuttgart, Germany
-
References
- 1 Safatle-Ribeiro AV, Baba ER, Iriya K. et al. Double-balloon endoscopy reveals sea-blue histiocytosis affecting the small bowel (with video). Gastrointest Endosc 2010; 72: 1266
- 2 Sharma P, Kar R, Dutta S. et al. Niemann-Pick disease, type B with TRAP-positive storage cells and secondary sea blue histiocytosis. Eur J Histochem 2009; 53: e22
- 3 Invernizzi R. Images from the haematologica atlas of hematologic cytology: sea-blue histiocytosis. Haematologica 2021; 106: 1
- 4 Anaut MB, Montero JA. Sea-blue histiocytosis. Ann Pathol 2024; 44: 150-151
- 5 Bermejo N, Prieto J, Arcos MJ. Sea-blue histiocytosis in bone marrow of a patient with chronic thrombocytopenia. Acta Haematol 2015; 133: 277-278