|Year : 2021 | Volume
| Issue : 1 | Page : 96-98
Masson hemangioma in a brachial artery aneurysm: A case report and short review
Manujaya Godakandage, Nalaka Gunawansa
Department of Vascular and Transplant Surgery, National Institute of Nephrology, Dialysis and Transplantation, Colombo, Sri Lanka
|Date of Submission||09-Apr-2020|
|Date of Decision||14-Apr-2020|
|Date of Acceptance||18-Apr-2020|
|Date of Web Publication||20-Feb-2021|
Department of Vascular and Transplant Surgery, National Institute of Nephrology, Dialysis and Transplantation, Colombo
Source of Support: None, Conflict of Interest: None
Masson hemangioma is a rare benign vascular tumor characterized by intravascular papillary endothelial hyperplasia (IPEH). Numerous case reports have described lesions in the digits, neck, oral cavity, and intracranially. IPEH involving primary brachial artery aneurysm is extremely rare, with only a handful of reported cases. We present a 43-year-old female with a true distal brachial artery aneurysm who presented with a painful pulsatile mass in the antecubital region. She underwent aneurysm resection and end-to-end repair with a saphenous vein graft. Histology confirmed the diagnosis of IPEH with characteristic features of papillary hyperplasia within the intravascular thrombus.
Keywords: Brachial artery aneurysm, intravascular papillary endothelial hyperplasia, Masson hemangioma
|How to cite this article:|
Godakandage M, Gunawansa N. Masson hemangioma in a brachial artery aneurysm: A case report and short review. Indian J Vasc Endovasc Surg 2021;8:96-8
| Introduction|| |
Intravascular papillary endothelial hyperplasia (IPEH), also known as Masson hemangioma, is a rare benign tumor of the vascular tree, accounting for 2%–4% of all soft tissue vascular tumors., Numerous case reports have emerged with reported IPEH affecting the vasculature in relation to digits, neck, oral mucosa, trunk, or intracranial vessels. IPEH in the brachial artery is extremely rare with only a handful of cases in the literature. Preoperative diagnosis is often difficult and requires a high degree of clinical suspicion. Surgical repair is often required in arterial IPEH, not only to relieve the associated symptoms but also to confirm the diagnosis and prevent complications.
| Case Report|| |
This index patient was a 43-year-old homemaker, presenting with a painful lump over the right cubital fossa, lasting over 2 months. The pain and the swelling were both progressive and causing significant discomfort in her dominant hand. There was no history of blunt or penetrating trauma to the region preceding the onset of symptoms. She was afebrile and there was no history of local or systemic sepsis. There were no other comorbidities.
On examination, there was a 4 cm × 2.5 cm firm lump in the antecubital region with an expansile pulsation. Both the radial and ulnar pulses were palpable and equal in volume to the contralateral side. There was neither distal sensory or motor impairment nor any evidence of distal ischemia.
A color duplex ultrasonography (DUS) was performed and showed a well-circumscribed aneurysmal segment of the distal brachial artery with intact flow and partial luminal thrombosis [Figure 1]. A computerized tomographic angiogram (CTA) was done [Figure 2] and conformed the above findings to pristine proximal and distal arterial anatomy. Her blood biochemistry including inflammatory markers was all within normal limits.
|Figure 1: Duplex ultrasound image showing the aneurysmal lesion with partial thrombosis|
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|Figure 2: Computerized tomographic angiogram of the right arm showing the distal brachial artery aneurysm with partial luminal occlusion|
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She underwent an open surgical exploration under general anesthesia. Proximal control was achieved at the distal brachial artery above the aneurysmal segment, while distal control was achieved individually at the origin of the radial and ulnar arteries. The aneurysmal segment was dissected and demonstrated just proximal to the brachial bifurcation [Figure 3]. The aneurysm segment was excised with a 2 cm proximal clearance and 1 cm distal clearance, leaving a common stem at the origins of the radial and ulnar arteries. Arterial continuity was achieved with an interposition vein graft using a reversed greater saphenous vein harvested from the contralateral leg. The postoperative period was uneventful, and she recovered with excellent distal perfusion having palpable radial and ulnar pulses. At the time of reporting (4 months postsurgery), she remains well, asymptomatic, and with no evidence of recurrence.
Histopathological examination of the resected arterial segment confirmed IPEH (Masson hemangioma) with intravascular papillary hyperplasia and an organized thrombus [Figure 4] and [Figure 5].
|Figure 4: Histology; ingrowth of the endothelium along the contours of the thrombus|
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| Discussion|| |
IPEH was first described by Pierre Masson in 1923, who observed the lesion in relation to hemorrhoids. Since then, it has been described in different locations of the body, with three distinct varieties.
- Primary IPEH, the most common variety, occurs de novo as part of pathologically dilated vessels such as arterial or venous aneurysm, similar to what was observed in this index patient. In this context, patients typically present with a short history of a distinct vascular lump and need to be differentiated from primary vascular tumors such as angiosarcoma
- Secondary IPEH occurs in relation to a preexisting dilated vascular lesion such as a varix, hemangioma, or arteriovenous malformation. These are long-standing lesions, and often, IPEH is identified as an incidental finding after surgical excision
- Finally, an extremely rare extravascular form of IPEH has been described in long-standing organized hematomas.
The exact pathophysiology of IPEH is poorly understood. The initial descriptions described IPEH as a true neoplastic lesion with degeneration and secondary thrombosis. However, more recent studies have described it as a mere benign “reactive” outgrowth in relation to organizing thrombi. Lesions are primarily described in relation to veins while those occurring in the arterial tree are less frequent. Histological diagnosis depends on the demonstration of exuberant endothelial proliferation, numerous papillary projections, and encroachment of the fibrin thrombus [Figures 4 and 5]. In contrast to the usual appearance and behavior of an organizing thrombus, IPEH demonstrates tumor-like growth with gradual expansion over time and recurrence if incompletely excised.
Clinical presentation depends on the location. The clinical presentation of Masson haemangiomata depends on their anatomical location. When it's located in the limb extremities, neck or oral mucosa, the commonest presentation is with the obvious mass, pain and discomfort. Intracranial lesions may act as space-occupying lesion, presenting with headache or seizures. A moderate female preponderance has been observed among the reported cases, sometimes linked to possible hormonal stimulation with the demonstration of increased estrogen and progesterone receptors in excised lesions.
Primary IPEH occurring in the brachial artery aneurysms is extremely rare. Due to the location and associated symptoms, these need definitive treatment by complete excision followed by reconstruction. In the index patient, diagnosis of a true brachial artery aneurysm was obtained after clinical examination and imaging. DUS is often inconclusive with the images of a well-circumscribed hypervascular lesion, while CTA is more informative in terms of anatomy and arterial continuity as observed in this patient.
Surgical excision is the preferred gold standard in the treatment of similar lesions occurring in the extremity arteries. It allows complete excision for histological confirmation as well as avoidance of recurrence. Arterial continuity is typically achieved by interposition vein graft as was done in this patient.
| Conclusion|| |
IPEH is a rare benign vascular lesion generally reported in relation to venous malformations. Arterial involvement is sporadic and rare, mainly in the digits, oral cavity, and intracranial arteries. Only two reported cases were found of IPEH in the brachial artery aneurysms., An aneurysmal growth with fairly rapid progression should be viewed with extreme caution. Clinical evaluation, biochemical tests, and imaging help in differentiating from pseudoaneurysm, mycotic aneurysm, and angiosarcomas. Definitive treatment involves complete excision and reconstruction with histological confirmation after excision. The long-term prognosis is excellent, and the recurrence is rare.
Explicit informed written consent has been obtained from the patient regarding the academic publication of this article with relevant details and images.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]