We report the case of a 34-year-old immunosuppressed woman who presented with severe
malabsorption and weight loss. Her history was remarkable for systemic lupus erythematosus
(SLE) complicated by antiphospholipid syndrome, lupus nephritis, and recurrent myocardial
pericarditis; she had been treated with combined intensive immunosuppressive regimens.
Her antitransglutaminase antibodies were negative and a flare-up of SLE was excluded.
Esophagogastroduodenoscopy revealed remarkable atrophy of the duodenal mucosa, which
appeared atypical for both celiac and Whipple disease. A small-bowel capsule endoscopy
was performed for disease staging and lesion characterization and showed severe diffuse
enteropathy with flattened and enlarged edematous villi and sparse hyperemic foci
from the duodenum to the middle portion of the ileum, compatible with severe diffuse
enteropathy ([Video 1]). A subsequent histopathological assessment on small-bowel mucosal biopsies showed
the presence of severe mucosal atrophy with amastigotes of Leishmania protozoa ([Fig. 1]), thereby confirming the diagnosis of visceral leishmaniasis. Owing to resistant
to amphotericin B, she was successfully treated with intravenous pentamidine.
Video 1 Small-bowel capsule endoscopy showing severe diffuse enteropathy with flattened and
enlarged edematous villi and hyperemic foci from the duodenum to the middle portion
of the ileum, compatible with intestinal leishmaniasis.
Fig. 1 Histology of the duodenal biopsy showing numerous macrophages in the lamina propria
of the duodenal mucosa, with abundant intracytoplasmic Leishmania amastigote parasites on: a hematoxylin and eosin (H&E) staining (magnification × 200); b Giemsa staining (× 200).
Visceral leishmaniasis is a life-threatening parasitic systemic disease caused by
species of the Leishmania genus, typically presenting with fever, hepatosplenomegaly, and pancytopenia [1]. Atypical presentations are also found in immunosuppressed and human immunodeficiency
virus (HIV)-infected patients [2]. In such cases, Leishmania parasites are usually detected in abnormal tissues, such as in the intestinal mucosa
[3]. The course is characterized by treatment failure and relapses, and the prognosis
is poor [2].
This case is interesting for several reasons. First, we describe an unusual case of
small-bowel leishmaniasis, which presented clinically with the typical symptoms of
severe malabsorption. In addition, for the first time we provide detailed video and
imaging documentation of a Leishmania-driven enteropathy to further support clinicians and endoscopists during their everyday
practice.
Endoscopy_UCTN_Code_CCL_1AC_2AG
Endoscopy E-Videos is a free access online section, reporting on interesting cases and new techniques
in gastroenterological endoscopy. All papers include a high
quality video and all contributions are
freely accessible online.
This section has its own submission
website at
https://mc.manuscriptcentral.com/e-videos
