Jejunal hemorrhage due to hemolymphangioma successfully detected and controlled by double-balloon enteroscopy.

Lymphangiomas are benign, often subclinical, neoplasms, which can develop in the digestive tracts. Hemorrhagic jejunal tumors are relatively rare and diagnostic challenge. We report herein a case of hemorrhagic jejunal hemolymphangioma successfully diagnosed and treated by double-balloon enteroscopy.

CASE PRESENTATION

A 74‐year‐old woman receiving an anticoagulant for atrial fibrillation was admitted to our hospital because of persistent abdominal pain and melena. Laboratory test results indicated severe anemia (hemoglobin, 3.0 g/dL). Upper gastrointestinal endoscopy and colonoscopy were performed, but no hemorrhagic lesion was identified. However, as tarry enteric fluid flowing out from ileocecal valve was observed in the colonoscopic examination, we speculated that the bleeding point existed at small intestine. Double‐balloon enteroscopy was performed, and as was expected, a small (ø6 mm) polypoid lesion with active bleeding was found at the jejunum (Figure 1A). The severe hemorrhage disturbed detailed observation. Endoscopic clipping was immediately done for hemostasis (Figure 1B). Subsequently, partial jejunectomy was conducted to prevent re‐bleeding. The surgical specimen had a tiny hemorrhagic lesion (Figure 2A), which histologically consisted of aggregation of dilated lympho‐vascular channels in the subepithelial region (Figure 2B). The final pathologic diagnosis was jejunal hemolymphangioma. To date, no recurrence of melena has been reported, and her hematological data have been improving (hemoglobin, 8.9 g/dL).

FIGURE 1

Representative images of double‐balloon enteroscopy. (A) A small polypoid lesion with active bleeding is seen (arrowheads). (B) Endoscopic clipping successfully stopped bleeding

FIGURE 2

Pathologic findings of the surgical specimen. (A) A small hemorrhagic polypoid lesion was confirmed in macroscopic observation (arrowheads). In a cut surface, the hemorrhagic change is located only at the apex of the lesion (inset). (B) A microscopic image of the apex portion shows aggregation of dilated vessels filled with lymphatic fluid and blood accompanied by extravasation of erythrocytes. (Hematoxylin‐eosin stain, ×40 magnification)

DISCUSSION AND CONCLUSION

Intestinal lymphangiomas/hemolymphangiomas are relatively rare benign neoplasms and mostly subclinical. Bleeding is an important manifestation in usually ≥20 mm tumors. , Our case suggests that lymphangiomas, even if small, can cause life‐threatening gastrointestinal hemorrhage.

CONFLICT OF INTEREST

None.

AUTHOR CONTRIBUTIONS

Y.Ito was responsible for writing the initial draft of the manuscript. Y.Ikura was responsible for conception, design, drafting, image modification, and finalizing the manuscript. KK was responsible for surgery and image modification. TO was responsible for design and finalizing. All authors read and approved the final manuscript.

ETHICAL APPROVAL

Informed consent for publication and related images has been obtained from the patient.

CONSENT

Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.