Segmental xanthomatosis of the small intestine. A case report and review of the literature

Delacruz, Victor; Takahashi, Hidenori; Nishida, Seigo; Tzakis, Andreas G.; Ruiz, Phillip

doi:10.1016/j.humpath.2008.05.016

Cited by 11 publications

(13 citation statements)

References 4 publications

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“…The etiologies of intestinal xanthomatosis not associated with predisposing conditions such as hyperlipidemia or lymphoproliferative lesions have not been settled and one of the theories related to its development states that an insult generates destruction of cells in the mucosa or submucosa with subsequent ingestion of lipid-containing debris by histiocytes which then persist as foamy cells [ 16 ]. Gastrointestinal xanthomas are rare and the stomach is the most frequent described location [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

“…Review of the literature about intestinal xanthomatosis shows that this entity is predominantly found incidentally during endoscopy; however, among the symptomatic cases, some presented with obstruction and strictures/stenosis that caused dysmotility of the intestinal musculature [ 6 ]. Also some cases had presented as small bowel pseudotumors [ 16 ]; the reported symptomatic patients by Delacruz et al had either hyperlipidemia or lymphoproliferative disorders [ 4 ]. Neither of these conditions was found in this case.…”

Section: Discussionmentioning

confidence: 99%

“…About 5.5% present with strangulation [ 3 ], making surgery the essential treatment to prevent further complications. Xanthomatosis is characterized by accumulation of lipid-laden, foamy macrophages resulting in nodule formation and commonly involving the skin [ 4 ]. In the gastrointestinal tract, stomach is the most frequent location; however, cases involving esophagus [ 5 ], small bowel [ 6 , 7 ], ileocecal valve [ 8 ], colon [ 9 – 12 ] and rectum [ 13 ], tendons, bladder, and prostate have been described [ 14 ].…”

Section: Introductionmentioning

confidence: 99%

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Small Bowel Obstruction due to Intestinal Xanthomatosis

Barrera-Herrera

Arias

Rodríguez‐Urrego

et al. 2015

Case Reports in Pathology

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Vast majority of bowel obstruction is due to postoperative adhesions, malignancy, intestinal inflammatory disease, and hernias; however, knowledge of other uncommon causes is critical to establish a prompt treatment and decrease mortality. Xanthomatosis is produced by accumulation of cholesterol-rich foamy macrophages. Intestinal xanthomatosis is an uncommon nonneoplastic lesion that may cause small bowel obstruction and several cases have been reported in the English literature as obstruction in the jejunum. We report a case of small intestinal xanthomatosis occurring in a 51-year-old female who presented with one day of copious vomiting and intermittent abdominal pain. Radiologic images revealed jejunal loop thickening and inflammatory changes suggestive of foreign body obstruction, diagnostic laparoscopy found two strictures at the jejunum, and a pathologic examination confirmed a segmental small bowel xanthomatosis. This case illustrates that obstruction even without predisposing factors such as hyperlipidemia or lymphoproliferative disorders.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Small Bowel Obstruction due to Intestinal Xanthomatosis

Barrera-Herrera

Arias

Rodríguez‐Urrego

et al. 2015

Case Reports in Pathology

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“…They rarely occur in the small intestine, colon or esophagus [7]. Outside the GI tract, xanthomas are more frequent in soft tissues and skin.…”

Section: Introductionmentioning

confidence: 99%

Non-gastric Gastrointestinal Xanthomas: Case Series and Literature Review

Arco

Sánchez

Aceñero

2016

JGLD

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Gastrointestinal xanthomas are infrequent non-neoplastic lesions characterized by the accumulation of foam cells in the lamina propria. They are commonly seen in association with dyslipidemia, chemotherapy or radiotherapy, and infections in immunosuppressed patients. However, no clear connection to hyperlipidemia has been found. They occur more frequently in the stomach, and are very rare in the small bowel and esophagus. We identified all cases of non-gastric xanthoma or xanthomatosis reported in the English literature by searching the PubMed database and retrospectively reviewed the clinical, endoscopic, and histopathologic features of the 11 cases of non-gastric gastrointestinal xanthomas diagnosed in our hospital. Nine lesions were located in the large bowel, one in the duodenum and one in the esophagus. All xanthomas were small (<5 mm) sessile polyps except the esophageal xanthoma, which measured 13 mm. Two cases in the large bowel and the case in the small bowel were multiple. Most patients with large bowel xanthomas had hypercholesterolemia, unlike esophageal and small bowel cases. The esophageal lesion occurred in a patient with a history of partial fundoplication due to gastroesophageal reflux disease and the small bowel case was associated to chronic atrophic gastritis with intense activity. In our search of the English literature, we found 19 cases of xanthoma or xanthomatosis in the esophagus, 13 cases in the small bowel and 61 cases in the large bowel. In conclusion, gastrointestinal xanthomas, other than the gastric ones, are rare, and are usually incidental findings. Abbreviations: AIDS: Acquired immunodeficiency syndrome; CESD: Cholesterol ester storage disease; ChT: Chemotherapy; CMV: Cytomegalovirus; GERD: Gastroesophageal reflux disease; HCL: Hypercholesterolemia; HCV: Hepatitis C virus; HPV: Human papilloma virus; MAI: Mycobacterium avium intracellulare; PCR: Polymerase chain reaction; RT: Radiotherapy.

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“…The histology of MCGs in the present patient showed abundant cholesterol crystals in addition to foamy histiocytes and inflammatory cells. It was different from that of xanthogranulomatosis 11 . Furthermore the patient did not have hyperlipidemia.…”

Section: Discussionmentioning

confidence: 62%

Multiple endocrine neoplasia type 1‐associated cystic pancreatic endocrine neoplasia and multifocal cholesterol granulomas

Kimura¹,

Komuro

Uchino

et al. 2010

Pathology International

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A novel combination of tumors was found in a 68 year-old female with Multiple Endocrine Neoplasia type-1 (MEN 1) that included a cystic pancreatic endocrine neoplasm (CPEN), a pituitary adenoma, and multifocal cholesterol granulomas (MCGs) in the breast, pleura, and the extremities. The pancreatic tumor displayed a single central locule surrounded by a thin rim of neoplastic parenchyma. The tumor showed heterogeneity in the architecture that included glandular, trabecular and solid patterns. The tumor cells of the pancreas were immunohistochemically positive for both endocrine and pancreatic acinar markers including chromogranin A, synaptophysin, glucagon, lipase, and reg protein. Electron microscopy revealed that there were numerous smaller dense-cored neurosecretory granules, larger zymogen-like granules and microvilli on the apical side of the tumor cells. The pancreatic tumor was diagnosed as CPEN with acinar cell features. Analysis of the DNA extracted from the tissues revealed that there is a MEN1 germline mutation in exon 10 codon 527, and somatic mutation in exon 2 codon 32 in the pancreatic tumor, and one base pair deletion in exon 2 codon 79 in the pituitary adenoma. Here, we report the case and discuss possible pathogenesis of CPEN and MCGs in a patient with MEN 1.

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Segmental xanthomatosis of the small intestine. A case report and review of the literature

Cited by 11 publications

References 4 publications

Small Bowel Obstruction due to Intestinal Xanthomatosis

Small Bowel Obstruction due to Intestinal Xanthomatosis

Non-gastric Gastrointestinal Xanthomas: Case Series and Literature Review

Multiple endocrine neoplasia type 1‐associated cystic pancreatic endocrine neoplasia and multifocal cholesterol granulomas

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