Appendiceal hemorrhage: An uncommon cause of lower gastrointestinal tract bleeding and intraluminal contrast extravasation.

A 32-year-old man with no medical history was admitted to our hospital for a sudden onset of hematochezia in the past 3 days. The patient had neither abdominal pain, vomiting, fever, nor weight loss, and he did not take any medicine like corticosteroid or nonsteroid anti-inflammatory drugs recently. On examination, the patient was awake with a Glasgow coma scale of 15 points, there were no signs of anemia, and his blood pressure was 110/70 mmHg. The complete blood count showed a slight decrease in hemoglobin levels (114 g/dL) and white blood cell count (9.63 g/L), and an average platelet count (190 g/L). The patient underwent a colonoscopy, highlighting bright red blood flowing along the colonic folds from the ileocecal angle to the rectum. The root of the appendix showed active bleeding without any signs of ulcers or warts ( Fig. 1 ). No ulcers, polyps, or diverticula were detected along the colonic framework. Fig 1 Colonoscopy image showing the bleeding root of the appendix (arrow). Fig 1 Colonoscopy image showing the bleeding root of the appendix (arrow). Computed tomography of the abdomen with intravenous contrast injection was performed the day after the colonoscopy, revealing extravasation of contrast material in the appendix (with no oral or rectal contrast administration). In addition, no appendiceal wall thickening or peri-cecal fatty stranding was found ( Figs. 2 A and B). The cecum wall was regularly thickened, mainly in the submucosal layer. The post-contrast image showed enhancement of the mucosal layer, consistent with chronic inflammation ( Fig 2 B; arrow). Fig 2 Postcontrast computed tomography image on the axial plane (A) and coronal plane (B) showing intraluminal contrast extravasation at the root of the appendix (arrow). Fig 2 Postcontrast computed tomography image on the axial plane (A) and coronal plane (B) showing intraluminal contrast extravasation at the root of the appendix (arrow). The patient subsequently underwent an endoscopic appendectomy. The surgical report documented that the small intestine, colon, and appendix appeared normal. A 6.0 cm in-length appendix with a typical GI tract layer appearance was noted, with a small bleeding ulcer node in the lumen of the root ( Fig 3 ). The histopathological result revealed erosion of the mucosa with hemorrhage. No granuloma formation or vascular lesion was documented ( Fig 4 ). The postoperative follow-up was uneventful, and the patient was discharged 2 days later. A routine checkup after 2 months detected no recurrence of hematochezia. Fig 3 Laparoscopic appendectomy image showing a normal appendix with a bleeding ulcer at the root portion (arrow). Fig 3 Fig 4 (A) Pathological examination revealed focal sloughing of the mucosa with hemorrhage (arrow), H&E, magnification × 10. (B, C) Mild infiltration of polymorphonuclear lymphocytes (white arrows), H&E, magnification × 40 (B), magnification × 10 (C). Fig 4 Laparoscopic appendectomy image showing a normal appendix with a bleeding ulcer at the root portion (arrow). (A) Pathological examination revealed focal sloughing of the mucosa with hemorrhage (arrow), H&E, magnification × 10. (B, C) Mild infiltration of polymorphonuclear lymphocytes (white arrows), H&E, magnification × 40 (B), magnification × 10 (C).

Our institution does not require ethical approval for reporting individual cases or case series. Written informed consent was obtained from the patient(s) for their anonymized information to be published in this article.

Informed consent for patient information to be published in this article was obtained.

Le TD and Nguyen DH contributed equally to this article as first authorship. Le TD and Nguyen DH: Case file retrieval and case summary preparation. Nguyen DH and Nguyen MD: preparation of manuscript and editing. All authors read and approved the final manuscript.

Our patient was successfully treated by surgical resection. This article highlights the importance of appendiceal assessment in lower GI tract hemorrhage, especially in young patients. Combining endoscopy and CT can improve the efficacy of appendiceal hemorrhage diagnosis.

Lower GI tract bleeding is uncommon, with an annual prevalence of 20.5 in 100,000, and often occurs in older male patients with average age ranges from 63 to 77 years. The colorectal region is the bleeding source in 80% of cases [4] . The many causes include diverticulitis, hemorrhoids, neoplasm, vascular dysplasia, colonic polyps, and ischemic colitis; infection is present in the vast majority of cases [3] . Appendiceal hemorrhage is an infrequent cause of lower GI tract bleeding. It is believed to relate to Crohn's disease, appendicitis, intussusception, vascular dysplasia, neoplasm, endometriosis, or focal sloughing of the mucosa. Table 1 illustrates the origin of 13 appendiceal hemorrhage cases. Hematochezia dominated, and abdominal pain was reported in 2 cases. Cases in women were less frequent than in men and young, and middle-aged patients were more common than in the elderly. Table 1 Information from the 13 published articles about appendiceal bleeding. Table 1 Year Age/Sex Clinical symptoms Definitive diagnosis Reference 2017 46/Male Abdominal pain Appendicitis [7] 2017 33/Male Abdominal pain Diverticulitis of the appendix [8] 2016 72/Male Hematochezia Vascular malformation [9] 2016 22/Male Rectal bleeding Granulomatous appendicitis [10] 2015 68/Male Hematochezia Dieulafoy lesion of the appendix [11] 2014 44/Male Hematochezia Diverticulitis [12] 2014 51/Male Hematochezia Dieulafoy lesion of the appendix [13] 2013 71/Male Hematochezia Ulcerated mucosa of the appendix [14] 2013 41/Male Hematochezia Non-appendiceal vascular hyperplasia [15] 2011 25/Male Hematochezia Focal sloughing of the mucosa [5] 2010 42/Male Hematochezia Focal sloughing of the mucosa [6] 2001 76/Female Hematochezia Vascular malformation [9] 1985 32/ Female Hematochezia Ulcerated mucosa of the appendix [16] Information from the 13 published articles about appendiceal bleeding. Focal sloughing of the mucosa is not a typical presentation. This paper is the third report of appendiceal hemorrhage with this cause. In 2011, Chiang et al. [5] reported an appendiceal hemorrhage in a 25-year-old man. Multislice CT was used to diagnose, and a good outcome was documented. The histopathologic result showed focal sloughing of the mucosa [5] . In 2010,Baek et al. [6] reported a 42-year-old man with appendiceal hemorrhage. The active bleeding point was detected at the tip of the appendix on the CT scan. An endoscopic appendectomy was performed, and no bleeding was reported in the postoperative examination. Microscopic examination revealed sloughing mucosa, focal inflammation, and no submucosal vascular malformation [6] . The advent of multi-slice CT has facilitated GI tract bleeding evaluation, especially with non-invasive, prompt, and accurate diagnosis (88.5%) [17] . Combining colonic endoscopy and abdominal CT has been proven to enhance diagnostic accuracy. Furthermore, multi-slice CT is crucial in pinpointing the bleeding source [9] . The treatment modality is personalized based on disease type and clinical status. Although endoscopic hemostasis can be useful in lower GI tract bleeding, no publication has reported successful endoscopic hemostasis in appendiceal hemorrhage. Surgery was the modality of choice for almost all focal appendiceal lesions. Extended resection, including cecectomy, ileostomy, or hemicolectomy, has been reported for hemostasis. Previous report revealed their experiences with temporary hemostasis in appendiceal dysplasia using an angiography (coil) intervention. Unfortunately, hemorrhage occurred after 2 weeks, and surgical resection was performed [9] .

Lower gastrointestinal (GI) tract bleeding is defined as hemorrhage originating from the GI tract segment below the Treitz ligament, which is usually life-threatening [1] . Various diagnostic modalities have been used to diagnose appropriately, including angiography, multidetector computed tomography (MDCT), and colonoscopy. However, making an accurate diagnosis with further interventions is still tricky. Despite several advents in radiology and colonoscopy modalities and techniques, pinpointing the source of bleeding remains a significant challenge in daily clinical practice, especially in severe or nonactive bleeding. The origin cannot be identified in an estimated 10% of lower GI tract bleeding cases [2] . The typical causes include vascular disease, Crohn's, neoplasm, inflammation, hemorrhoids, and ischemic colitis [3] . Lower GI tract bleeds due to appendiceal hemorrhage are rare and only occasionally reported. This paper reports an appendiceal hemorrhage in a young male patient. He was diagnosed using an abdominal computed tomography (CT) scan and GI tract endoscopy.

Data and materials used and/or analyzed during the current study are available from the corresponding author on reasonable request.