Multiple small bowel cavernous hemangiomas complicated with intussusception presenting with intestinal obstruction: rare case report from Sudan and literature review

Abstract

Introduction:

Multiple cavernous hemangiomas (MCH) of the small intestine are a rare condition of the small bowel, with intussusception being an even rarer complication.

Case presentation:

We report the first documented case of MCH from Sudan. An 11-year-old Sudanese woman with a long history of unexplained anemia presented with acute abdominal pain, vomiting, and absolute constipation, indicative of a mechanical bowel obstruction supported by X-ray findings of the abdomen. Failing conservative management, she underwent an emergent laparotomy. Intraoperatively, an ileoileal intussusception with multiple hemangiomas as the lead point, causing proximal bowel dilatation and ischemia, was discovered. Segmental resection of the ischemic portion of the small bowel and wedge resections of the largest hemangiomas were performed. Histology confirmed the diagnosis of cavernous hemangioma. The patient had an uncomplicated postoperative course and remained well during follow-up. AI was used to improve writing and organization.

Discussion:

This case highlights the diagnostic challenge of small bowel hemangiomas, particularly when presenting with rare complications like intussusception, although in this case it presented with typical and some occasional features of hemangioma.

Conclusion:

While surgical resection remains the gold standard for symptomatic cases of MCH, this report underscores the importance of considering this rare condition in patients with unexplained chronic anemia, acute abdominal symptoms, or both, to ensure timely and effective management.

Introduction

Multiple cavernous hemangiomas (MCH) of the small intestine is an extremely rare pathology, furthermore complicating with intussusception being a much rarer condition^[1,2^].

Hemangiomas are benign tumors of the vascular endothelium, representing 0.05% of all gastrointestinal neoplasms^[1,3^], with 7–10% located in the small intestine^[4^]. Jejunum is the most common site followed by the ileum, then the colon, and can occur at any age, with no sex preference^[4,5^]. Cavernous subtype is the most common type and usually is a solitary lesion^[6,7^].

It may be asymptomatic, or present commonly with acute lower gastrointestinal bleeding that can cause anemia on long-term, or abdominal pain and rare complications include intussusception, intestinal obstruction and perforation^[8^].

HIGHLIGHTS

• This study reports the first case of multiple cavernous hemangiomas of the small intestine complicated with intussusception in Sudan.

• Can present with the classic, but often overlooked, symptom of chronic anemia.

• Confirmation of cavernous hemangioma via histology is the gold standard and surgical resection is the definitive treatment.

Intussusception in pediatrics is usually idiopathic and less likely to be secondary to a pathological lead point like masses and diverticula, with more than 90% of cases being ileocolic^[9,10^]. The gold standard treatment of symptomatic cases is surgical resection^[11^].

To the best of our knowledge, this is the first reported case in Sudan of Intussusception secondary to MCH presenting with intestinal obstruction. In Sudan, especially after the 15 April 2023 conflict, resources are limited in many areas and hence early diagnosis of such cases and management. The work has been reported in line with the SCARE criteria 2025^[12^].

Case presentation

History and physical examination

An 11-year-old Sudanese woman, weighing about 28 kg, with a long history of anemia presented to our emergency department with sudden onset abdominal pain accompanied by excessive vomiting for 1 day. She experienced moderate abdominal pain that was localized in the epigastric and umbilical areas, which was non-radiating and non-migratory. Regarding the vomiting, she had four large episodes of non-bilious, non-bloody vomiting. accompanied by absolute constipation for the same duration. No history of hematemesis, hematochezia, or weight loss. Past medical history was significant for recurrent hospitalizations due to anemia without a known cause, but there was no history of similar conditions or surgical operations.

On clinical examination, she appeared ill, pale, and not jaundiced. Initial vital signs indicated slight tachycardia (109 beats per minute), respiratory rate 30 per minute, with normal body temperature 36.2°C. Abdominal examinations revealed localized tenderness in the epigastrium, but there were no signs of peritonitis, palpable masses, or organomegaly. Auscultation revealed high-pitched, hyperactive bowel sound and no vascular bruits.

Diagnostic evaluation

Laboratory investigations on admission revealed normal white blood cell (WBC) with left shifting (WBC 4.1 k/µL; segmented neutrophils 83 %) and significant anemia (Hb 7.1 g/dL); all other routine laboratory parameters were within normal limits. Plain abdominal radiography (supine and upright views) (Fig. 1) showed dilated small bowel loops and multiple air-fluid levels, which are Indicative of mechanical obstruction. Abdominopelvic ultrasound revealed dilated small and large bowel with fluid-filled dilated lumen, along with intestinal obstruction with possible volvulus.

Figure 1.

Plain abdominal X-ray: (A) Upright showing air fluid level; (B) Supine showing dilated bowel loops.

Treatment

The patient was initially managed conservatively with intravenous fluids and nasogastric tube decompression, analgesia, and broad-spectrum intravenous antibiotics, but unfortunately, the patient failed to improve clinically with this conservative treatment, necessitating urgent surgical intervention. The patient underwent emergent laparotomy during the exploration, an ileoileal intussusception, causing obstruction and dilatation of the proximal bowel, with a hemangioma as a lead point (Fig. 2). Further exploration disclosed multiple (about 10) hemangiomas and evidence of multiple ischemic bowels. After reduction, segmental resection of about 70 cm of the ischemic portion was performed (Fig. 3). In addition, wedge resections of the largest hemangiomas and primary end-to-end anastomosis are performed. The patient received about 10 units of blood according to his age and weight. Of notice that laparoscopy and endoscopy were not attempted due to limited resources.

Figure 2.

Intraoperative findings after exploration and reduction of the intussuscepted bowel. (A) Dilated proximal bowel with evidence of abnormal masses in the ileum; (B) Bowel after reduction and relive of the obstruction.

Figure 3.

The resected portion of the ileum containing the lesions of cavernous hemangioma. (A) The resected ileal segment with evidence of abnormal masses and bowel ischemia; (B) The resected part in the surgical dish.

Histological findings of the resected specimen reported: macroscopically reddish small bowel segment, with two donut lesions (aggregate size 2.5 × 2 × 1 cm). Cross-section showed edematous red-brown mucosa and submucosal well-circumscribed red firm mass measured 1.7 × 1.5 × 1 cm. Microscopic findings included loss of mucosa and ulceration, with necrosis and hyalinization of muscularis propria and dilated, congested blood vessels; submucosal nodules showed benign neoplasm composed of kyanized dilated blood vessels lined by benign endothelial cells, no granuloma, dysplasia or malignancy seen; all considered as intestinal ischemia with viable surgical margin cavernous hemangioma.

Follow-up

The patient’s postoperative course was uncomplicated and was discharged on the seventh postoperative day. Close follow-up for 1 month revealed a smooth recovery with resolution of abdominal symptoms and no further bleeding episodes. Subsequent telephone follow-up confirmed the patient remained well without recurrence of similar complaints.

Discussion

This is the first documented case of small bowel MCH in a Sudanese patient although cases of MCH that led to intussusception and intestinal obstruction represent rare presentations^[11^] (Table 1).

Table 1.

Literature review of pediatric cases of small bowel hemangioma.

	Author	Publication year	Age	Sex	Clinical presentation	Lesion size(s)	Histological/Morphological type	Anatomical location	Intervention performed	Outcome and follow-up duration
1	Copple et al [19]	1961	5 years	M	Persistent anemia, epistaxis, melena, hematemesis, systolic murmur, syncopal attacks	1.5 cm (2 lesions)	Dilated venous channels	Jejunum	Surgical resection and primary bowel anastomosis	No further GI bleeding at 3 months
2	Jackson et al [20]	1967	7 years	F	Nausea, vomiting, diarrhea, edema, abdominal distention, pale, hepatomegaly	2–3 cm (several), 3–4 mm (innumerable)	Diffuse, infiltrating, cavernous hemangioma	Distal jejunum, terminal ileum	Surgical resection	Asymptomatic, edema-free at 14 months
3	Elefant et al [13]	1970	Newborn	M	Bile-stained vomiting, abdominal distention, no meconium	NA	Arteriocapillary with calcification and necrosis-hamartoma	Small intestine	Jejunoileostomy, enterotomy	Symptom-free at 2.5 years
4	Magnano et al [21]	2005	13 years	M	Fatigue, anemia, positive fecal occult blood	2 cm (1 lesion)	Cavernous	Ileum	Surgical resection	No bleeding at 1-year follow-up
5	Jones et al [22]	2007	10 & 7 years	F, F	Recurrent abdominal pain, anemia, collapse, melaena	2 cm (2 lesions)	Vascular malformation, cavernous hemangioma	Jejunum	Surgical resection with anastomosis	Normal hematinics at 6 months
6	Jarvi et al [23]	2008	3 months	F	Melena, failure to thrive, anemia	NA	Infantile hemangiomatosis	Small bowel	Thalidomide + somatostatin therapy	Asymptomatic at 2 years
7	Pinho et al [24]	2008	9 years	F	Melena, anemia, fatigue, nausea	2 × 2.5 cm (1 lesion)	Cavernous	Ileum	Surgical resection	Asymptomatic post-op
8	Khanna et al [25]	2010	11 years	M	Pallor, fatigue, melena, severe anemia	NA	NA	Jejunum & jejuno-ileal junction	Surgical resection	Thriving at 6 months
9	Rao et al [26]	2010	5 weeks	F	Non-bilious emesis, constipation, failure to thrive	0.5–2.2 cm (multiple)	Diffuse hemangiomatosis	Ileum	Resection + hemicolectomy	Discharged, follow-up at 2 weeks
10	Abdul Aziz et al [8]	2011	6 years	F	Abdominal pain, vomiting, palpable masses	15 cm (1 lesion)	Cavernous	Ileum	Surgical resection	Symptom-free at 6 months
11	Pera et al [27]	2012	16 years	M	Fatigue, palpitations, anemia	4.3 × 4.2 cm (1 lesion)	Cavernous	Jejunum	Surgical resection	No GI bleeding at 1 year
12	Shukri et al [28]	2013	27 days	M	Fever, distension, bilious vomiting	NA	Capillary	Ileum	Resection with anastomosis	Discharged on day 10
13	Bae et al [29]	2015	13 years	M	Dizziness, fatigue, recurrent anemia	4.2 × 5.2 cm (1 lesion)	Cavernous	Jejunum	Surgical excision	Hb 14.6 g/dL at 3 years
14	Coleman et al [30]	2018	2 years	F	Severe anemia, pallor, black stools	4.5 × 2 cm (1 lesion)	Benign with fibrosis, calcification	Small bowel	Surgical resection	No anemia recurrence at 3 months
15	Krick et al [31]	2018	7 weeks	F	Bloody stools, anemia, feeding intolerance	NA	Capillary infantile hemangioma	Small bowel wall & mesentery	Laparotomy, omentectomy, propranolol	Asymptomatic at 15 months
16	Chen et al [32]	2020	12 years	M	Abdominal pain, fever, peritonitis	5 × 3 × 2 cm (1 lesion)	NA	Small intestine	Laparoscopic enucleation	Discharged on day 7
17	Helal et al [33]	2024	3 months	F	Rectal bleeding, crying, pallor	4.6 × 2.1 × 1.5 cm (1 lesion)	Mixed hemangioma	Jejunum	Segmental resection + anastomosis	Discharged after 7 days
18	Kong et al [34]	2024	21 months	F	Blood in stool, anemia, fever	6 × 2.5 × 1.2 cm (1 lesion)	Cavernous	Jejunum	Single-site laparoscopic resection	Hb 129 g/L at 3 months
19	Singh et al [35]	2024	3 years	M	Weakness, transfusions, anemia	4.9 × 2.2 × 3.8 cm (solitary)	Cavernous (submucosal)	Ileum	Resection + anastomosis	NA
20	Vlad et al [36]	2024	6.5 years	F	Severe anemia, failure to thrive, black stools	NA	Cavernous	Ileum and jejunum	Laparotomy + tumor removal	Normal Hb & iron, no bleeding for 1 year
21	Current study	2025	11 years		Intestinal obstruction, intussusception, and chronic anemia	10 lesions	Cavernous	Ileum	Surgical resection	Discharged on day 7. Follow-up revealed no complications.
						Size: NA

Age of this patient was 11, while the most common age-group according to literature is 3–7 years old with possibility to occur at any age; as early as newborns and as late at 87 years old cases were reported in literature^[13,14^]. The most common presenting symptoms were gastrointestinal bleeding (melena, hematochezia) and anemia, followed by abdominal pain and obstruction^[8^]; however, this case presented with symptoms of anemia for long time, and due to the delay in diagnosis of anemia cause, the patient developed late complications of hemangioma and presented with abdominal pain and intestinal obstruction. This finding aligns with prior literature emphasizing that small bowel hemangiomas should be considered in cases of unexplained chronic anemia or occult bleeding, particularly after negative upper and lower endoscopies. In 10% of hemangioma cases, they were discovered as incidental finding during imaging or surgery^[11^].

The majority of lesions were cavernous hemangiomas, as the case in this report, but in contrast to literature where the jejunum is the most affected site, the lesions in the current case were located in the ileum resulting in ileoileal intussusception. Lesions size varied significantly (0.5–15 cm), with larger lesions more likely to cause obstruction or intussusception. Interestingly, diffuse hemangiomatosis was reported in pediatric cases, often necessitating extensive resections. Other features of the tumors in this case include size, number, and macroscopic appearance, which were consistent with literature. Although most cases are solitary, multifocal lesions were found in many cases as in this report^[6,7,11,15^].

Diagnosing small bowel hemangiomas is notoriously difficult due to their rarity, variable presentation, and lack of pathognomonic features and the limitations of conventional endoscopy, the submucosal location of these lesions^[15^].

Many cases were diagnosed intraoperatively – as in this case – or via CT/MRI enterography according to literature^[11,14^]; however, clinicians should consider this diagnosis in atypical cases, such as pediatric patients with failure to thrive or elderly individuals with vague abdominal complaints or unexplained chronic anemia.

Diagnosis is frequently delayed, with misdiagnosis in 50% of initial presentations. Undetected intussusception risks bowel ischemia, perforation, and mortality up to 32%^[16^].

Intussusception is the second most common cause of pediatric bowel obstruction, which is mostly idiopathic and <10% are due to pathologic lead point, with Meckel’s diverticulum being the most common cause rather than tumors and other masses^[16,17^].

Key differentials include inflammatory bowel disease, intestinal tuberculosis, angiodysplasia, and neoplasms like lymphoma or gastrointestinal stromal tumor^[18^]. Hemangiomas must be considered in all patients undergoing evaluation for unexplained anemia or gastrointestinal bleeding, especially when standard endoscopic and imaging modalities fail to identify a source.

Surgical resection remains the gold standard for symptomatic hemangiomas. As highlighted in the literature, these benign lesions lack metastatic potential, and local excision of the affected segment is curative^[1,3^]. While propranolol has demonstrated success in isolated cases of neonatal gastric hemangiomas surgery is unequivocally preferred for life-threatening complications like obstruction, perforation, or hemorrhage.

For MCH as in this case, the extent of resection requires careful balancing: radical excision must be weighed against the risk of short bowel syndrome. As emphasized in literature^[11,14^], conservative resection that removes symptomatic segments while preserving bowel length is critical to prevent nutritional deficiencies.

Laparoscopic exploration offers significant advantages, such as enabling thorough abdominal evaluation, targeted resection, and intraoperative enteroscopy to localize bleeding sites^[11^] This approach reduces blood loss, postoperative pain, wound complications, and hospitalization duration. In our case, laparotomy was necessitated by severe bowel distension; however, laparoscopic techniques should be prioritized when feasible.

In this case, close follow-up in the first month and next months showed complete resolution of symptoms without recurrence which is typical for cases of gastrointestinal hemangiomas; however, mortality was reported in one case (Ralston, 1960)^[37^] due to postoperative pneumonia emphasizing the importance of perioperative care in high-risk patients.

Based on a literature review of 20 pediatric cases (under 18 years old) of small bowel hemangioma, a total of 541 reports were initially identified via a PubMed search (Table 1). These were screened down to 62 papers for data extraction, revealing key findings about the condition. The patient cohort comprised 11 women and 9 men, with ages ranging from newborn to 16 years. The most common clinical presentations were anemia (15 cases) and gastrointestinal bleeding (10 cases). Cavernous hemangioma was the most frequent histological type (9 cases), predominantly located in the jejunum (12 cases). Surgical resection was the primary treatment, used in 18 of the 20 cases, and all reported cases had a positive outcome with symptom resolution. This analysis provides a concise summary of the current literature, noting that the new case represents the 21st documented pediatric case of small bowel hemangioma.

Conclusion

Small bowel hemangiomas, though rare, are an important cause of gastrointestinal bleeding and anemia. Surgical resection remains the gold standard, but minimally invasive and endoscopic techniques are increasingly viable. Early recognition in highly suspicious cases is crucial to early diagnosis and prevents serious complications, where one limitation of this case is the delay in the diagnosis.

Ethical approval

Ethical approval for the study was obtained from the ethical committee at Atbara Teaching Hospital, Atbara River Nile state, Sudan. The study was conducted in accordance with the principles of the Declaration of Helsinki. Oral informed consent was obtained from the parents for publication and the images while ensuring confidentiality and anonymization of the data.

Consent

Written informed consent was obtained from the patient’s parents for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Sources of funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Author contributions

J.A.: Conceptualization, writing – original draft preparation; S.S.A., A.M., and M.E.: Writing – Reviewing and Editing; A.M.: Data curation. M.E.: Data curation; J.A.: Data interpretation; S.S.A.: Submission. All authors read and approved the final manuscript.

Conflicts of interest disclosure

Authors declare no competing interests.

Registration of research studies

http://www.chictr.org.cn/index.aspx.

Guarantor

Jaber Amin.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Data availability statement

The data of this report are available upon reasonable request from the corresponding author.

Declaration of generative AI

Generative AI was only used to improve language and readability.

Declarations

Not applicable.