Gastric Bloom: Flower-like Lesions of Isolated Gastrointestinal Langerhans Cell Histiocytosis
Year: 2025
Type: article
Study design: Case Report
Abstract: CASE REPORT A 64-year-old man with tobacco use disorder underwent routine low-dose chest computed tomography for lung cancer screening, which revealed diffuse esophageal thickening. He was referred to the gastroenterology clinic and remained asymptomatic aside from occasional heartburn. Esophagogastroduodenoscopy revealed a normal esophagus but multiple small, flower-like lesions in the gastric body and fundus (Figure 1). Biopsies showed large mononuclear cells expanding the lamina propria, accompanied by eosinophils and small lymphocytes. The lesional cells exhibited oval to irregular nuclei with occasional nuclear grooves (Figure 1). Immunohistochemistry demonstrated positivity for S100 and strong CD1a expression, and a B-raf proto-oncogene, serine/threonine kinase (BRAF) V600E mutation was detected, confirming the diagnosis of Langerhans cell histiocytosis (Figure 1). Positron emission tomography/computed tomography demonstrated disease confined to the stomach. Given the absence of symptoms and lack of multisystem involvement, a surveillance strategy with repeat positron emission tomography/computed tomography every 6 months was pursued in collaboration with oncology. Langerhans cell histiocytosis is rare in adults, and isolated gastrointestinal involvement is especially uncommon.1,2 Although prior reports have described variable endoscopic findings, the subtle appearance in our case illustrates how easily such lesions may be overlooked and emphasizes the importance of thorough endoscopic evaluation.3,4 The case also highlights the role of multidisciplinary collaboration in managing rare gastrointestinal manifestations of systemic disease.Figure 1.: Endoscopic and histopathologic findings. (A, B) Esophagogastroduodenoscopy revealed multiple small, flower-like lesions (arrows) in the gastric body and fundus, characterized by a central depression surrounded by petal-like mucosal elevation, shown under high-definition white-light endoscopy system in (A) and with narrow-band imaging for optical enhancement in (B). (C, D) Biopsies showed infiltration of Langerhans cells with irregular, grooved nuclei (arrows), accompanied by eosinophils and lymphocytes expanding the lamina propria (hematoxylin and eosin, ×400) in (C), and demonstrated strong CD1a expression by immunohistochemistry (×200) in (D).DISCLOSURES Author contributions: J. Alp, S. Goyal, and TJ Davie contributed equally to this work, as outlined below, and share first authorship. J. Alp contributed to the acquisition of data, drafted the manuscript, and participated in editing and finalizing the manuscript. S. Goyal contributed to the acquisition of data, drafted the manuscript, and participated in editing and finalizing the manuscript. TJ Davie contributed to the acquisition of data, drafted the manuscript, and participated in editing and finalizing the manuscript. AM Cioc made contributions to the conception and design of the work by editing the histopathology section, providing microscopy photos, and writing the figure legends for the histopathology images. D. Guerrero Vinsard supervised case management and manuscript development, revised the manuscript critically, and provided senior mentorship. All authors approved the final version to be published and agree to be accountable for all aspects of the work. D. Guerrero Vinsard is the article guarantor. Financial disclosure: None to report. Informed consent was obtained for this case report.
Source: ACG Case Reports Journal
Authors: Jameel Alp, Samarth Goyal, Timothy J. Davie, Adina M. Cioc, Daniela Guerrero Vinsard
Language: English
Subfield: Physiology
Field: Medicine
Domain: Health Sciences
Open Access status: diamond
OpenAlex ID: W4417039298