Appendiceal bleeding caused by vascular malformation: A case report

Abstract

BACKGROUND

Acute lower gastrointestinal bleeding (LGIB) is a common occurrence in clinical practice. However, appendiceal bleeding is an extremely rare condition that can easily be overlooked and misdiagnosed. The preoperative detection of appendiceal bleeding often poses challenges due to the lack of related guidelines and consensus, resulting in controversial treatment approaches.

CASE SUMMARY

We presented a case of a 33-year-old female who complained of hematochezia that had lasted for 1 d. Colonoscopy revealed continuous bleeding in the appendiceal orifice. A laparoscopic appendectomy was performed immediately, and a pulsating blood vessel was observed in the mesangium of the appendix, accordingly, active bleeding into the appendicular lumen was considered. Pathological examination revealed numerous hyperplastic vessels in the appendiceal mucosa and dilated capillary vessels.

CONCLUSION

The preoperative detection of appendiceal bleeding is often challenging, colonoscopy is extremely important, bowel preparation is not routinely recommended for patients with acute LGIB or only low-dose bowel preparation is recommended. Laparoscopic appendectomy is the most appropriate treatment for appendiceal bleeding.

Key Words: Lower gastrointestinal bleeding, Appendiceal bleeding, Colonoscopy, Vascular malformation, Laparoscopic appendectomy, Case report

Core Tip: Acute lower gastrointestinal bleeding (LGIB) is common, however, appendiceal bleeding is extremely rare. The diagnosis of appendiceal bleeding is challenging because of its rarity and lack of related guidelines and consensus. Colonoscopy is extremely important, and bowel preparation is not routinely recommended for patients with acute LGIB. Laparoscopic appendectomy is the most appropriate treatment for appendiceal bleeding.

INTRODUCTION

Lower gastrointestinal bleeding (LGIB) is defined as hemorrhage originating from the gastrointestinal tract segment below the Treitz ligament[1]. Various diagnostic modalities, including digital subtraction angiography (DSA), computed tomography angiography (CTA) and colonoscopy, are used for appropriate diagnosis. However, pinpointing the source of bleeding remains a significant challenge[2]. Only a few guidelines and a consensus on LGIB, especially, no major guidelines for appendiceal bleeding. Appendiceal bleeding is extremely rare, and easily missed and misdiagnosed[3]. Here, we report a case of appendiceal bleeding in which the clinical manifestation was hemafecia, and the cause of the appendiceal bleeding was vascular malformation.

CASE PRESENTATION

Chief complaints

A 33-year-old female presented to our hospital with hematochezia that had lasted for 1 d.

History of present illness

The patient reported a prior episode of passing approximately 300 mL of bloody stool before presenting to the hospital. There were no accompanying gastrointestinal symptoms, such as nausea, vomiting, or abdominal pain. Following the administration of 4 L of polyethylene glycol (PEG)-based bowel preparation, the patient experienced another episode of bloody stool, amounting to approximately 300 mL. The patient denied any previous history of hematochezia.

History of past illness

The patient had no history of acute or chronic infectious diseases, heart disease, hypertension or diabetes, or surgery.

Personal and family history

The patient had not recently taken any medicine, such as corticosteroids or nonsteroidal anti-inflammatory drugs. There was no family history of similar cases or bleeding disorders.

Physical examination

There were no signs of anemia, her blood pressure was 100/54 mmHg, and her heart rate was 96 beats per minute. The patient did not have any pathological signs.

Laboratory examinations

Her hemoglobin level was 134 g/L (normal range: 130-175 g/L), her leucocyte count was 15.87 × 10⁹/L, and her percentage of neutrophils was 83.4%. Other routine relevant examinations, such as platelet counts and coagulation function tests, yielded normal findings.

Imaging examinations

An emergency colonoscopy was performed, and the endoscope was extended to the terminal ileum. Continuous bleeding was observed at the appendiceal orifice (Figure 1A). In addition, a contrast-enhanced abdominal computed tomography scan revealed structural disorder in the ileocecal area, and the appendix was not clear (Figure 1B).

Figure 1 Colonoscopy and contrast-enhanced abdominal computed tomography scan. A: Continuous bleeding was observed at the orifice of the appendix; B: Structural disorder in the ileocecal area, and the appendix was not clear.

FINAL DIAGNOSIS

Appendiceal bleeding.

TREATMENT

An emergency laparoscopic appendectomy was performed. During the surgery, the appendix was 90 mm × 5 mm in size and the appendix was swollen 3-5 cm from the appendicular root (Figure 2A). A pulsating blood vessel was observed in the mesangium of the appendix (Figure 2B). Active bleeding into the appendicular lumen was considered, and a large number of blood clots were observed in the lumen. Pathologically, a large number of hyperplastic vessels were observed in the appendix mucosa and capillary vessels were dilated (Figure 3).

Figure 2 Specimens of the appendix and intraoperative images. A: The appendix was swollen 3-5 cm from the appendicular root; B: A pulsating blood vessel could be observed in the mesangium of the appendix.

Figure 3 The pathology of the appendix. A and B: A large number of hyperplastic vessels were observed in the appendix mucosa and capillary vessels were dilated.

OUTCOME AND FOLLOW-UP

The patient had no recurrent hematochezia or melena, and was discharged from the hospital 5 d after the surgery. One month later, no evidence of rebleeding was observed.

DISCUSSION

Acute LGIB is not an uncommon condition, with an incidence of approximately 0.02%, and it is more common in men and older people. Approximately 80% of cases are of colorectal origin, and appendiceal bleeding is extremely rare[4,5]. A search of the PubMed/MEDLINE database for literature published between 2010 and August 2023 regarding “appendiceal bleeding” or “appendiceal hemorrhage” identified 16 patients (Table 1). Appendiceal bleeding was more common in males than in females (male:female = 15:1), and the average age of the included patients was 54.75 years (range: 25–90 years). The causes of appendiceal bleeding mainly include vascular malformation, ulcers, inflammation, neoplasms, mucosal erosion and Dieulafoy’s lesions. In addition, the causes of appendiceal bleeding in 40% of patients are unknown.

Table 1 Case of appendiceal bleeding.

Case	Age (yr)/sex	Techniques	Treatment	Complications	Pathology	Ref.
1	32/male	Colonoscopy	Endoscopic appendectomy	None	Appendiceal ulcer	[2]
2	90/male	Colonoscopy	Laparoscopic appendectomy	None	Unknown	[3]
3	70/male	Colonoscopy	Endoscopic clipping	None	None	[4]
4	25/male	Colonoscopy	Appendectomy	None	Appendiceal mucosal erosion	[5]
5	54/male	Colonoscopy	Appendectomy	None	Unknown	[10]
6	32/male	Colonoscopy	Laparoscopic appendectomy	None	Dieulafoy’s lesion	[10]
7	72/male	Colonoscopy	Laparoscopic appendectomy and cecum wedge resection	None	Vascular malformation	[11]
8	73/male	Colonoscopy	Appendectomy	None	Appendiceal mucosal erosion	[12]
9	32/male	Colonoscopy	Appendectomy	None	Appendiceal ulcer	[13]
10	72/male	Colonoscopy	Right hemicolectomy	None	Appendiceal mucinous adenocarcinoma	[14]
11	49/male	Colonoscopy	Appendectomy with partial cecal resection	None	Acute appendicitis	[15]
12	88/male	Colonoscopy	Appendectomy	None	Low-grade mucinous neoplasm of appendix	[16]
13	42/male	Colonoscopy	Laparoscopic appendectomy	None	Appendiceal mucosal erosion	[17]
14	44/male	Colonoscopy	Colonoscopic clipping	None	None	[18]
15	34/female	Colonoscopy	Colonoscopic clipping	None	None	[19]
16	67/male	Angiogram	Embolization the appendiceal artery	Acute appendicitis	None	[20]
Our case	33/female	Colonoscopy	Laparoscopic appendectomy	None	Vascular malformation

The diagnosis of appendiceal bleeding mainly includes CTA, DSA and colonoscopy. CTA is suitable for patients with active bleeding (bleeding rate ≥ 0.3 mL/min). Additionally, DSA is affected by the bleeding rate. When the bleeding rate is greater than 0.5 mL/min, the detection rate reaches 50%–72%, but if the bleeding rate is lower than 0.5 mL/min, the detection rate decreases significantly[6]. Colonoscopy plays an irreplaceable role for LGIB, at least to the terminal ileum[7].

No consensus exists regarding on bowel preparation before emergency colonoscopy for LGIB. Some scholars believe that adequate bowel preparation and clear exposure under the scope are the basis for discovering and treating all lesions, and recommend 4-6 L of PEG-based bowel preparation; in addition, split-dose preparation and/or the use of low-volume preparations can also be considered[8]. However, some scholars believe that oral laxatives can aggravate gastrointestinal bleeding and shock[9]. According to our case, the patient experienced massive hematocheia again after receiving oral laxatives. We believe that bowel preparation is not routinely recommended for patients with acute LGIB or that only low-dose bowel preparation is recommended for the following reasons: (1) Aggravation of gastrointestinal bleeding; (2) The equipment used for colonoscopy is more advanced, and which can be observed while washing, therefore, it is helpful to find and treat the lesion site; and (3) The blood in the intestinal lumen itself has a cathartic effect.

No standard treatment is available for appendiceal bleeding, and it has been reported that local spraying of hemostatic drugs can successfully stop bleeding. Hemostasis can also be achieved by endoscopic clamping of part of the appendiceal orifice or appendiceal artery embolization[4,10,11]. The choice for most scholars is appendectomy[3]. In our case, the site of bleeding in the patient's appendix was approximately 3 cm from the orifice, and the appendix was slender. The hemostatic effect of spraying hemostatic drugs alone may be poor. Partial occlusion of the appendiceal orifice with colonoscopic clipping or appendiceal artery embolization increases the risk of acute appendicitis, and there is the possibility of rebleeding[11]. Therefore, Laparoscopic appendectomy is the most appropriate treatment for appendiceal bleeding, and colonoscopy is performed during the operation when necessary.

CONCLUSION

The number of reports on appendiceal bleeding is limited, and there is a lack of corresponding guidelines and consensuses on standardizing the clinical diagnosis and treatment. We eagerly await more large-scale research to provide evidence for evidence-based medicine in clinical practice.