Early and accurate diagnosis and selection of appropriate treatment plans are crucial for patients with gastrointestinal hemangiomas

Abstract

Gastrointestinal hemangioma (GIH) is clinically rare, accounting for 7%-10% of benign gastrointestinal tumors and 0.5% of systemic hemangiomas. GIH can occur as either solitary or multiple lesions, with gastrointestinal bleeding as a significant clinical manifestation. Understanding the clinical and endoscopic features of GIH is essential for improving diagnostic accuracy, particularly through endoscopy and selective arteriography, which are highly effective in diagnosing GIH and preventing misdiagnosis and inappropriate treatment. Upon confirmed diagnosis, it is essential to thoroughly evaluate the patient's condition to determine the most suitable treatment modality—whether surgical, endoscopic, or minimally invasive intervention. The minimally invasive interventional partial embolization therapy using polyvinyl alcohol particles, proposed and implemented by Pospisilova et al, has achieved excellent clinical outcomes. This approach reduces surgical trauma and the inherent risks of traditional surgical treatments.

Key Words: Hemangioma; Gastrointestinal hemangioma; Gastrointestinal hemorrhage; Minimally invasive interventional treatment; Selective embolization

Core Tip: For patients presenting with unexplained gastrointestinal bleeding, recurrent melena, and chronic anemia, gastrointestinal hemangiomas should be highly suspected. Active endoscopic and other diagnostic examinations are essential to confirm the lesion and prevent misdiagnosis or inappropriate treatment. Following a confirmed diagnosis, a comprehensive assessment of the patient's condition is necessary to determine the most suitable treatment method, whether surgical intervention, endoscopic therapy, or minimally invasive interventional therapy.

TO THE EDITOR

Hemangioma is a common soft tissue tumor, frequently found in the head and neck region[1]. However, gastrointestinal hemangioma (GIH) is relatively rare. With the increasing use of gastrointestinal endoscopy, the detection rate of GIH has risen annually[2], particularly in cases of asymptomatic or small lesions. The main symptoms of GIH are gastrointestinal bleeding and obstruction. Chronic intermittent gastrointestinal bleeding may begin in childhood and progressively worsen with age. In cases where GIH is complicated by infection, systemic symptoms such as fever and elevated white blood cell count may occur. Rupture of a hemangioma can cause significant bleeding, which, in severe cases, may lead to shock or even death. Tu et al[3] conducted a retrospective study comparing endoscopic and surgical treatments for GIH, highlighting the importance of endoscopy for accurate diagnosis. Despite numerous case reports, misdiagnosis remains common, and the optimal treatment methods for GIH patients continue to be a topic of debate.

In the latest issue of the World Journal of Gastrointestinal Surgery, an article titled "Anorectal hemangioma, a rare cause of lower gastrointestinal bleeding, treated with selective embolization: A case report" has garnered significant interest. This clinical case report by Pospisilova et al[4] provides a valuable reference for the safe and effective diagnosis and treatment of intestinal hemangioma. Pospisilova et al[4] described a 21-year-old male patient who had experienced intermittent painless rectal bleeding since childhood. Unfortunately, he was initially misdiagnosed with ulcerative colitis (UC) and underwent long-term treatment with azathioprine and mesalazine, yielding poor therapeutic outcomes. His condition continued to deteriorate due to intermittent rectal bleeding, leading to anemia and necessitating a blood transfusion. It was not until age 18 that he was referred to a specialized gastroenterology center, where a comparative colonoscopy finally revealed the correct diagnosis. This case highlights the challenges in diagnosing GIH, often resulting from insufficient awareness and limited diagnostic consideration among clinicians. Therefore, vigilance in cases of gastrointestinal bleeding, coupled with the early use of diagnostic methods such as endoscopy and selective angiography, is essential for improving the accurate diagnosis of GIH and avoiding prolonged mismanagement.

Incidence and characteristics of GIH

GIH was first described in 1839[5-8]. Clinically, GIH is rare, accounting for 7%-10% of benign gastrointestinal tumors and 0.5% of systemic hemangiomas. GIH can occur in any part of the gastrointestinal tract but is most commonly found in the jejunum, followed by the ileum and colon. It typically appears as a blue-purple, sessile or polypoid lesion, often originating from the submucosal vascular plexus and sometimes involving the muscular layer or even the serosa. Most lesions are smaller than 2 cm in diameter, although rectal lesions may be larger. GIH is generally classified into capillary hemangioma, cavernous hemangioma, and mixed hemangioma. Its clinical manifestation is non-specific and similar to other causes of gastrointestinal bleeding, with primary symptoms being gastrointestinal bleeding and obstruction. GIHs are typically solitary, but multiple lesions are common in patients with Blue Rubber Bleb Nevus Syndrome[9,10], who present with cutaneous hemangiomas from birth. Although isolated cutaneous hemangiomas are frequently observed, caution is warranted when symptoms such as hemorrhagic anemia develop.

Active investigation should be conducted to confirm the lesion and further guide clinical treatment in patients with a high suspicion of GIH

GIH, though rare, is an important cause of lower gastrointestinal bleeding. Despite numerous case reports, misdiagnosis remains a concern[5-7,9]. Amarapurkar et al[11] reported three cases in which rectal hemangiomas were misdiagnosed as UC, resulting in prolonged incorrect treatment. This not only adversely affected treatment outcomes but also worsened patient conditions and increased their financial burden. These cases underscore the importance of accurate diagnostic methods. Effective diagnostic methods for GIH include endoscopy, abdominal plain radiography, barium enema, and selective angiography. Although endoscopic biopsy is the gold standard for diagnosis, it should be avoided in suspected hemangioma lesions due to the risk of severe bleeding[12,13]. Endoscopy can reveal characteristic features such as visible mucosal vasculature, compressible nodules, and extensive submucosal hemangioma lesions with a bluish tint. The extent of proximal involvement can be determined by assessing the range of mucosal abnormalities; however, reports indicate that hemangiomas may infiltrate the deeper serosal layer through connective tissue in the muscular layer[14], making the assessment of deeper lesions challenging. In summary, in cases of unexplained gastrointestinal bleeding, recurrent melena, and chronic anemia, GIH should be highly suspected. Active endoscopic and other diagnostic evaluations are crucial to confirm the lesions and guide further clinical treatment.

Treatment options for GIH are diverse and each carries risks, with alternative treatments showing promise

Intestinal hemangiomas do not resolve spontaneously and can lead to chronic hemorrhagic anemia; therefore, lesion removal is recommended whenever possible[15]. With the widespread use of endoscopic examinations, small and asymptomatic GIHs are increasingly being detected. Endoscopic treatment methods, including endoscopic mucosal resection, endoscopic submucosal dissection, and endoscopic sclerotherapy, have proven effective in treating GIH[2,16-19]. If an endoscopic examination identifies polypoid lesions in the intestine, endoscopic resection or ablation may be performed[20]. However, these treatments carry risks, including uncontrolled gastrointestinal bleeding, intestinal perforation, and a high rate of recurrence[21]. Pharmaceutical treatments, such as oral propranolol, have been reported to successfully treat gastric hemangioma in neonates[22]. Nevertheless, for asymptomatic hemangiomas that present with life-threatening symptoms such as bleeding, intussusception, intestinal obstruction, and intestinal perforation, surgical resection remains the preferred treatment[20].

Surgical resection of the affected intestinal segment is currently considered the primary treatment for intestinal hemangioma[23]. Since hemangiomas are benign and do not metastasize to lymph nodes or distant tissues and organs, local resection is generally sufficient[20]. Sphincter-preserving procedures, such as rectal anterior resection and coloanal anastomosis, are preferred to maintain function. In some cases, thorough abdominal exploration is warranted due to the potential involvement of extracolonic organs[24], though this approach increases surgical risks and overall treatment costs.

For pedunculated GIH lesions, endoscopic resection offers advantages over surgical resection, although it carries higher risks of bleeding and perforation. Therefore, careful endoscopic approaches and effective bleeding prevention measures are crucial[25]. In a case reported by Pospisilova et al[4], the treatment plan took into account potential anemia exacerbation from gastrointestinal bleeding, the risks of fecal incontinence following surgical treatment, and the possibilities of ischemia and postoperative bowel stenosis caused by vascular embolization. Thus, a minimally invasive approach was proposed, involving mesenteric angiography and partial embolization of the rectal branch vessels using PVA particles. Following this treatment, the patient's gastrointestinal bleeding symptoms improved significantly, with endoscopy revealing only a small ischemic ulcer at the embolization site. Subsequent follow-up colonoscopy confirmed ulcer healing without bowel stenosis. This alternative treatment method proposed by the authors achieved excellent clinical outcomes, avoiding surgical trauma and the adverse quality-of-life impacts, psychological stress associated with colostomy, as well as unexpected risks of surgical treatment. However, further research is required to validate this method for treating GIHs of varying locations, numbers, extents, and depths.

CONCLUSION

GIH is a rare but significant cause of gastrointestinal bleeding, often presenting with non-specific symptoms that lead to frequent misdiagnosis. Severe cases of GIH can result in life-threatening symptoms, such as hemorrhagic shock, underscoring the importance of early diagnosis and appropriate treatment selection. Comparative studies on different treatment methods for GIH remain limited (Table 1), presenting a promising avenue for future research. Key areas for further investigation include: (1) Pathogenesis: A deeper understanding of GIH pathogenesis is essential. Future research should focus on the mechanisms, prevention, and treatment of hemangiomas, advancing both theoretical and applied approaches to improve diagnostic and therapeutic methods; (2) novel treatment methods: While current treatment methods include pharmaceutical treatment, photodynamic therapy, cryotherapy, embolization, and surgical intervention, exploring combinations with emerging methods, such as laser, radionuclide, and biological therapies, could improve cure rates and patients' quality of life; (3) personalized treatment plans: As medical research advances, there is growing potential for personalized treatment approaches, with therapy tailored to the patient's specific conditions, including hemangioma size, location, and associated symptoms; and (4) interdisciplinary collaboration: The study and treatment of hemangiomas intersect with multiple fields such as genetics, immunology, and structural biology. In summary, advancing GIH treatment will require increased interdisciplinary collaboration to develop innovative treatment methods and strategies.

Table 1 Comparison of clinical efficacy of different treatment methods.

Ref.	Year	Diagnosis	Number of cases	Examination(s)	Treatment method(s)	Clinical efficacy
Pospisilova et al[4]	2024	Anorectal vascular malformation	1	Colonoscopy, abdominal CT and pelvic MRI, mesenteric artery angiography	Selective angiography + embolization	Good
Shen et al[5]	2016	Colonic angioma	1	Abdominal plain film, barium enema, abdominal CT and MRI, colonoscopy	Laparotomy + low rectal anterior resection	Good
Ganesananthan et al[6]	2019	Vascular tumor of the rectum and sigmoid colon	1	Colonoscopy, abdominal CT and pelvic MRI, pelvic angiography	Conservative treatment	Poor
Neto et al[7]	2017	Anorectal vascular malformation	1	Colonoscopy, endoscopic capsule, abdominal CT	Surgical resection of the affected intestinal segment	Good
Zhou et al[16]	2021	Hemangioma, lymphangioma, and vascular lymphangioma	31	Colonoscopy and endoscopic ultrasound, abdominal CT, pathological biopsy	Selective endoscopic treatment or surgical treatment	Good
Aoyama et al[19]	2020	Small intestine vascular tumor	1	Dual balloon endoscopy	Endoscopic injection of polydocanol into the lesion	Good
Kong et al[20]	2024	Small intestine vascular tumor	1	Abdominal ultrasound and CT	Surgical resection of the affected intestinal segment	Good
Kaya et al[22]	2018	Gastric angioma	1	Gastroenteroscopy	Temporary regression after treatment with propranolol	Good
Peng et al[23]	2016	Mesenteric vascular tumor	1	Gastroscopy, small intestine capsule endoscopy, abdominal CT	Exploratory laparotomy + surgical resection of the affected small intestine and mesentery, followed by side-to-side anastomosis of the small intestine	Good

MRI: Magnetic resonance imaging; CT: Computed tomography.

ACKNOWLEDGEMENTS

The authors acknowledge and appreciate our colleagues for their valuable suggestions and assistance for this editorial