Abstract
Intramuscular hemangiomas (IMHs) are uncommon benign vascular lesions, which develop in skeletal muscle. Herein, two cases of IMHs involving the cervical scalene musculature are presented. Such lesions are exceedingly rare, and can be difficult to distinguish from vagal paragangliomas based on clinical presentation, exam, and radiography. Complete surgical excision is the treatment of choice, and was successful in our two index patients. While rare, IMHs of the scalene muscles should be considered in the differential diagnosis of deep neck space masses.
1
Introduction
Intramuscular hemangiomas (IMHs) are rare benign vascular lesions, which develop in skeletal muscle and make up < 1% of all reported hemangiomas . These benign tumors represent congenital vascular malformations that usually remain subclinical until sudden growth results in pain, a palpable mass, or raises cosmetic concern . IMHs have been reported in almost every skeletal muscle of the head and neck, with the masseter representing the most common location . However, to our knowledge, there have been only 2 previous reports of IMHs involving the scalene musculature . While accurate preoperative diagnosis of an IMH is often extremely challenging due to their rarity and non-specific clinical findings, IMHs of the scalene musculature, as seen in our case series, present a unique challenge as they present clinically and radiographically similar to the more common vagal paraganglioma (VPG).
We present 2 cases of IMHs of the scalene musculature highlighting the natural history, clinical and radiological presentation, and available treatment modalities. These data are further supplemented with a review of the literature.
2
Case series and review of the literature
2.1
Case 1
A 25-year-old female presented with a 5-year history of a left sided mid cervical neck mass, which she first noted during pregnancy. The mass had grown slowly over time, and resulted in intermittent discomfort. Computed tomography (CT) demonstrated a hypervascular mixed density well circumscribed mass displacing the internal jugular vein and carotid system anteriorly ( Fig. 1 A ). Magnetic resonance imaging and venography (MRA/V) demonstrated an enhancing mass with salt and pepper flow voids located between C2 and the level of the cricoid cartilage, which resulted in displacement of the internal and external carotid arteries anteriorly ( Fig. 1 B). Angiography demonstrated anterior displacement of the carotid vasculature, and a patent internal jugular vein ( Fig. 1 C).
Laboratory work up was negative for catecholamine secretion. Preliminary diagnosis of VPG was made and the patient was taken to the operating room. Intraoperatively, a 6 × 3.5 × 1.5 cm vascular mass was found associated with the scalene musculature separate from the vagus nerve and wide local resection was achieved. Histopathology revealed a mixed type IMH with vascular proliferation of capillaries and well-formed vessels infiltrating and splaying apart the native skeletal muscle fibers. ( Fig. 2 A , B).
2.2
Case 2
A 21-year-old male presented with a 7-month history of a painless mass in his left mid cervical neck. This had slowly grown, and was temporally related with the onset of intermittent headache, hypertension, and dysphagia. CT scan demonstrated an enhancing vascular tumor extending from the carotid bifurcation to the lower neck ( Fig. 3 A ). MRI demonstrated a left carotid space mass, which displaced the common carotid artery anteriorly ( Fig. 3 B) and MRA identified 2 feeding vessels including a branch of the left vertebral artery and a large branch originating from the left subclavian artery. Angiography demonstrated intense vascular blush and select vessel embolization was performed ( Fig. 3 C).
Laboratory evaluation was negative for catecholamine secretion and a preliminary diagnosis of VPG was made. The patient was taken to the operating room and a 4 × 3.5 × 2 cm mass was found intimately associated with the scalene musculature and anterior rami of C5 and C6. The tumor was excised en bloc and was consistent with a mixed IMH on histopathology containing both moderate sized vessels and areas of small capillaries and endothelial cells ( Fig. 2 C, D).
2.3
Literature review
Review of the international literature revealed two additional cases of IMH originating from the scalene musculature ( Table 1 ) among over 80 reported cases involving other head and neck subsites. Both cases occurred in men in their second decade of life and presented with a painless supraclavicular mass. In each, physical exam revealed a soft, horizontally mobile but vertically restricted supraclavicular mass without associated bruits, thrills, or skin involvement. Both were managed successfully with complete surgical excision, and histopathology revealed a mixed IMH in one patient and a capillary IMH in the other.
| Author, Year | Age/Sex | Symptoms | Location | Imaging | Treatment | Pathology |
|---|---|---|---|---|---|---|
| Scott, 1957 | 21/M | Painless, right supraclavicular mass × 3 mo | Posterior scalene | Normal plain film X-ray | Complete resection, significant bleeding | Mixed type IMH |
| Ferlito, 1980 | 25/M | Painless, left supraclavicular mass × 2 mo | Middle scalene | None | Complete resection, no complications | Capillary IMH |
| Van Abel, 2012 | 25/F | Uncomfortable, left mid cervical mass × 5 years, noted during pregnancy | Scalene NOS | CT, MRI, MRA/V, Angiography | Complete resection, no complications | Mixed type IMH |
| 21/M | Painless, left midcervical mass × 7 mo | Scalene NOS | CT, MRI, MRA/V, Angiography, Embolization | Complete resection, no complications | Mixed type IMH |
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