Successful Treatment of Extensive Infantile Hemangiomatosis of the Small Bowel in a 3‐month‐old With Thalidomide and Somatostatin Analog

Abstract

Tumors of the small intestine are rare, constituting <5% of all gastrointestinal neoplasms (1). Gastrointestinal hemangiomas account for only 0.05% of all intestinal neoplasms (2). Patients with gastrointestinal hemangiomatosis can present with abdominal pain, intermittent bleeding, and anemia of varying severity, and sometimes obstruction or intussusception (2). Small-bowel hemangiomatosisusuallyisdifficulttodiagnosebecauseofitsrelative rarity and the fact that the workup for gastrointestinal bleeding is initially focused on the stomach, duodenum, and colon. We present a rare case of a 3-month-old female with extensive infantile hemangiomatosis affecting two thirds of the small bowel managed medically with thalidomide and somatostatin analog bitherapy.

Intestinal hemangioma presenting as recurrent hematochezia in a 6-week-old male

“…Some cases have been paired ultrasound and CT scan [4], used laparoscopy [8], or used scintigraphy with technetium-99m-labeled red blood cells and angiography [9] to identify the source of hematochezia as a gastrointestinal hemangioma. We used a combination of different imaging modalities, primarily ultrasound and MRI, which prompted exploratory laparotomy and therefore leading to the diagnosis.…”

Diffuse infantile hemangiomatosis of the ileum presenting with multiple perforations: a case report and review of the literature

“…Intestinal hemangiomas are rare, affecting males more frequently than females, in a 1:3 ratio [2]. They undergo rapid proliferation for up to 10 months of age [1]. Most involute in childhood and are rarely symptomatic after the proliferative phase.…”

“…However, it is generally thought that they are angiogenically driven, most likely with an increase in production of angiogenic stimulator [16]. Recently, antiangiogenic agents such as thalidomide and somatostatin have been shown to have beneficial effects on halting proliferation of hemangiomas [1]. Interferon alpha is also rarely utilized with caution because of its high incidence of neurotoxicity.…”

Long-time octreotide in an adolescent with severe haemorrhagic gastrointestinal vascular malformation

“…Only coagulation factor supplementation may have some benefit 10. The feasibility of octreotide treatment, however, was shown in numerous studies, mostly in adults5 12–14 including one randomised study,11 which were recently reviewed in a meta-analysis 15. Patients with bleeding gastrointestinal vascular malformations responded significantly to treatment with octreotide 15.…”

“…This occasionally leads to transfusion dependency, and may even be life threatening 4 5. While haemangiomas might be a target of pharmacological therapies like steroids, interferon, some other antiangiogenetic substances, or, as recently described, β-sympatholytic agents,6 these drugs usually are ineffective in vascular malformations.…”