Lower gastrointestinal bleeding attributed to xanthogranulomatous appendicitis: A case report

Abstract

BACKGROUND

Acute appendicitis is an infrequent etiology of lower gastrointestinal bleeding (LGIB), with approximately 20 reported cases globally. To the best of our knowledge, xanthogranulomatous appendicitis (XGA) presenting with LGIB has not been described in the literature.

CASE SUMMARY

The patient was a 41-year-old man admitted for blood in stool for 1 day. He had previously been healthy. One month ago, the patient underwent a routine health check-up, which revealed a hemoglobin (HGB) level of 145 g/L. Following hematochezia, repeat laboratory testing showed a quick decrease in HGB level to 94 g/L. Emergency colonoscopy revealed dark red blood throughout the intestinal lumen, with visible fresh red bleeding emanating from the appendiceal lumen. Bleeding was intermittent. After copious irrigation with normal saline, observation for approximately 3 minutes found persistent fresh red bleeding from the appendiceal lumen, while no blood staining was observed in the terminal ileum. The patient underwent laparoscopic appendectomy, and postoperative pathology suggested XGA with mucosal ulceration and hemorrhage.

CONCLUSION

XGA represents an unusual etiology of LGIB.

Key Words: Lower gastrointestinal bleeding; Etiology; Xanthogranulomatous appendicitis; Prognosis; Case report

Core Tip: Lower gastrointestinal bleeding (LGIB) has diverse etiologies, yet identifying the source remains challenging in approximately 10% of cases. Appendiceal bleeding is a rare condition that often necessitates repeated colonoscopy for diagnosis. Appendicitis is a recognized cause of LGIB, but bleeding due to xanthogranulomatous appendicitis (XGA) has not been reported in the literature. We present possibly the first case of XGA presenting with LGIB, which was diagnosed endoscopically and confirmed surgically.

INTRODUCTION

Lower gastrointestinal bleeding (LGIB) is a frequent clinical challenge with a complex etiology. Common causes include tumors, vascular malformations, inflammatory bowel disease, ischemic bowel disease, hemorrhoids, and diverticulitis. Angiography, multislice spiral computed tomography (CT), and endoscopy are the primary diagnostic modalities in clinical practice. Despite advances in radiologic techniques and endoscopic methods, accurate identification of the bleeding site remains challenging in clinical practice, especially in cases of severe or occult hemorrhage. The cause of bleeding cannot be identified in approximately 10% of LGIB cases[1].

LGIB originating from the appendix is mostly documented in case reports. Because appendiceal bleeding is typically intermittent and slow, multiple colonoscopic examinations may be required to establish the diagnosis[2]. Repeated irrigation of the appendiceal orifice and lumen can enhance the detection rate[3,4]. LGIB caused by xanthogranulomatous appendicitis (XGA) is extremely rare, and to the best of our knowledge, no cases have been previously reported in the literature. This article reports a case of XGA with bleeding in a 41-year-old male patient, diagnosed via endoscopy and confirmed by surgery.

CASE PRESENTATION

Chief complaints

A 41-year-old Chinese male presented to the outpatient gastroenterology clinic with a 1-day history of hematochezia.

History of present illness

Symptoms began 1 day ago, characterized by the sudden passage of a large volume of bright red blood per rectum.

History of past illness

The patient’s medical history was unremarkable. One month ago, the patient underwent a routine health check-up, which revealed a hemoglobin (HGB) level of 145 g/L.

Personal and family history

The patient denied any family history of malignant tumors.

Physical examination

On physical examination, the vital signs were as follows: Body temperature, 36.3 °C; blood pressure, 107/60 mmHg; heart rate, 95 beats per minute; respiratory rate, 23 breaths per minute. The palpebral conjunctivae were pale. The abdomen was flat and soft, without visible distension or tenderness. Bowel sounds were active at 6 times per minute.

Laboratory examinations

The HGB level dropped quickly from 145 g/L 1 month previously to 94 g/L. The white blood cell count was normal at 5.2 × 10⁹/L. Serum tumor marker levels were normal (carcinoembryonic antigen, 3.2 ng/mL).

Imaging examinations

Emergency colonoscopy revealed dark red blood throughout the intestinal lumen, with visible fresh red bleeding emanating from the appendiceal lumen (Figure 1). Contrast-enhanced CT of the appendix showed mild luminal dilation (0.8 cm in diameter), localized wall thickening, and contrast enhancement on dynamic imaging. Gas was present within the appendiceal lumen, and the surrounding fat spaces were clear (Figure 2).

Figure 1 Emergency colonoscopy image. Colonoscopy revealed dark red blood throughout the intestinal lumen, with visible fresh red bleeding emanating from the appendiceal lumen.

Figure 2 Contrast-enhanced computed tomography of the appendix. A and B: Computed tomography demonstrated mild luminal dilation, localized wall thickening, and contrast enhancement on dynamic imaging; gas was present within the appendiceal lumen, and the surrounding fat spaces were clear.

FINAL DIAGNOSIS

XGA was identified as the underlying etiology of the patient’s hematochezia.

TREATMENT

Given the significant decrease in the HGB level and the limited endoscopic therapeutic options for appendiceal bleeding, the patient declined endoscopic retrograde appendicitis therapy. He underwent laparoscopic appendectomy (Figure 3). Postoperative pathology suggested vascular dilatation with congestion and bruising, accompanied by ferritin deposition and a peripheral multinucleated giant-cell reaction, as well as mucosal ulceration and hemorrhage, consistent with XGA (Figure 4).

Figure 3 Gross specimen after appendectomy.

Figure 4 Postoperative pathology. A-C: Histopathological analysis and haematoxylin and eosin staining. Postoperative pathology suggested vascular dilatation with congestion and bruising, accompanied by ferritin deposition and a peripheral multinucleated giant-cell reaction, as well as mucosal ulceration and hemorrhage, consistent with xanthogranulomatous appendicitis (hematoxylin-eosin staining, 40 ×).

OUTCOME AND FOLLOW-UP

Postoperatively, the patient recovered well and was discharged on postoperative day 3. During the 3-month follow-up, the patient remained asymptomatic without recurrent hematochezia.

DISCUSSION

Appendiceal bleeding, a rare cause of gastrointestinal bleeding, accounts for about 0.014% of LGIB cases[3]. Some cases present with acute appendiceal bleeding, but most are chronic and insidious[5]. The reported causes of appendiceal bleeding include appendicitis[6], diverticulum, angiodysplasia[7,8], inflammatory bowel disease[9], endometriosis[10], and Dieulafoy’s lesion[2], with appendicitis being the most common cause[11]. Appendiceal bleeding is self-limiting, often mild, and may stop spontaneously or resolve with non-specific medical therapy. This may partially explain why appendiceal bleeding is relatively rare, and its actual incidence might be underestimated[3].

XGA is a rare clinicopathologic manifestation. Histologically, it is characterized by bright yellow or golden yellow mass-like lesions, epithelioid histiocytes, and a layer of xanthomatous cells. Neutrophils and mononuclear inflammatory cells are seen in the background, interspersed with multinucleated macrophages containing foamy histiocytes (xanthomatous cells) mixed with varying numbers of other inflammatory cells[12,13]. It accounts for only 0.25%-0.64% of all appendicectomy cases[13] and is more commonly seen in delayed appendicectomy[14], as appendicectomy is the final treatment option. A literature review suggests that the long-term prognosis for these patients is favorable[13].

In the present case, vascular ectasia and congestion secondary to XGA resulted in mucosal ulceration and bleeding, which may represent the underlying etiology of the patient’s lower gastrointestinal hemorrhage.

CONCLUSION

Bleeding from XGA is clinically important as a rare cause of LGIB. It is crucial to confirm the diagnosis in a timely manner and adopt appropriate therapeutic measures.

ACKNOWLEDGEMENTS

We thank the anesthesia and nursing teams of the Endoscopy Center at Zibo Central Hospital for their valuable contributions.