Abscess formation within the biceps brachii muscle is a rare phenomenon, with only five cases reported in the English literature. These instances have been linked to various triggers, such as intramuscular injections, traumatic hematoma formation [1,2], and systemic conditions including tuberculosis [3], Gonococcal disease [4], and diabetes. Notably, one case involved a diabetic patient who presented with septic shock secondary to a seemingly spontaneous biceps abscess [5]. Additionally, intramuscular hemangiomas, rare benign vascular neoplasms, are infrequently considered in the differential diagnosis for musculoskeletal pain [6]. Intramuscular hemangiomas are elusive in nature and confusing in clinical presentation, especially in the upper extremities, where they are rare. Intramuscular hemangiomas, constituting less than 1% of all hemangiomas, are known for their challenging diagnostics [7]. Typically emerging in early adulthood, these vascular neoplasms present with nonspecific clinical symptoms, complicating early detection. Pain is a predominant symptom in 60% of cases and, in the absence of other symptoms, often leads to clinical misdiagnosis due to the absence of characteristic features like bruit, pulsation, or thrill [7]. An intramuscular hemangioma can remain asymptomatic for an extended period until sudden growth triggers pain or functional impairment [8]. In such cases, though pain initially prompted medical attention, significant limitations in elbow movement became the focus, leading to the discovery of extensive muscle involvement.
Intramuscular hemangiomas, an exceedingly rare subset of vascular neoplasms, are histologically categorized into four types based on vessel size: cavernous, capillary, venous, and mixed [9]. The mixed subtype, often associated with a prolonged history of symptoms, was identified in a unique case with a 1-year history of pain. Diagnostic imaging plays a crucial role in confirming the presence and characteristics of intramuscular hemangiomas. Plain radiographs may reveal increased soft-tissue density indicative of a mass, and the detection of phleboliths, although present in only 25% of cases, is considered highly specific for hemangiomas [10]. Ultrasound examination typically depicts intramuscular hemangiomas as hyperechoic structures, occasionally accompanied by dot-like and linear blood flow signals on Doppler flow imaging [11]. However, due to the low blood flow within hemangiomas, Doppler signals may not be consistently detected. In the reported case of intramuscular hemangioma coexisting with abscess formation in the biceps brachii, magnetic resonance imaging (MRI) with gadolinium enhancement emerged as the pivotal diagnostic modality. The MRI findings revealed an ill-defined enhancing lesion within the left lateral part of the biceps muscle, consistent with intramuscular hemangioma [12]. Simultaneously, an infectious abscess in the medial part of the biceps muscle was identified, highlighting the coexistence of these two distinct pathologies.
We report a rare case of intramuscular hemangioma with abscess formation of the biceps brachii with significant functional limitations of elbow extension and forearm pronation for which the patient underwent surgical excision. The removed mass was confirmed histopathologically as a mixed-type intramuscular hemangioma with an associated abscess, attributed to methicillin-sensitive staphylococcus aureus infection. This study was approved by the Institutional Review Board of Korea University Ansan Hospital (No. 2023AS0363). This study was conducted retrospectively, and the requirement for informed consent was waived.
CASE REPORT
A 56-year-old male presented with a 5-year history of intermittent pain in the flexor aspect of the right elbow joint. Over the previous 2 years, he had noticed swelling in the right cubital fossa, leading to restrictions in elbow extension and forearm pronation. No significant trauma to the right elbow was reported. There was no history of injection or other interventional treatment except an intravenous line related to phlebitis about 2 weeks prior to presentation. The patient had underlying conditions including diabetes mellitus and a history of gastric cancer. Physical examination revealed a painful intramuscular mass (40 mm×20 mm) proximal to the right cubital fossa. No bruit or thrill was detected, but he reported local heat sensation and swelling and redness of the overlying skin. While elbow flexion and forearm supination were within normal ranges, although limitations were observed in elbow extension and forearm pronation.
Conventional radiography of the right elbow displayed a soft tissue swelling with multiple diffuse phleboliths, suggesting a potential hemangioma (Fig. 1). MRI indicated a hypoechoic mass with dot-like and linear blood flow signals within the tumor and diffuse swelling and enhancement of the biceps muscle with a non-enhancing area in the medial part of the muscle (Fig. 2). These findings collectively led to the diagnosis of an intramuscular hemangioma with infectious myositis in the biceps brachii muscle. Surgical excision was performed using a flexor approach with a curvilinear incision under general anesthesia. Dissection aimed to achieve complete resection of the intramuscular hemangioma while preserving surrounding functional neurovascular structures. Intraoperatively, a 42 mm×20 mm×15 mm-pathologic mass with yellowish-gray drainage was identified within the distal muscle belly of the biceps brachii, extending to the biceps brachii tendon (Fig. 3). The mass appeared spongy and angiomatous with relatively ill-defined margins. Following complete resection (Fig. 4), elbow extension returned to the normal range without additional procedures. A short elbow plaster was applied for 2 weeks postoperatively, at which time the Hemovac drain was removed, and the patient initiated functional elbow exercises as physical therapy and intravenous antibiotics with cefazolin injection 1 g every 8 hours for the next 2 weeks. Before surgery, the patient's CRP level was significantly elevated at 25.43 mg/dL, indicating a severe inflammatory response. At 3 weeks postoperatively, the CRP level had returned to the normal range (0.32 mg/dL), reflecting the resolution of the infection. By 5 weeks after surgery, all functional limitations in the right elbow and forearm had resolved. Full elbow extension was achieved, and the ranges of motion in pronation and supination of the right forearm mirrored those of the contralateral side. No evidence of recurrence was observed during the 5-month follow-up period.
DISCUSSION
Intramuscular hemangiomas are exceptionally rare vascular tumors, with a prevalence less than 1% among all hemangiomas. The etiology of intramuscular hemangiomas remains unclear, with potential factors including congenital origin, minor trauma, or excessive muscle contraction [6,8]. These lesions typically occur in the lower extremities, and the case under discussion involving the biceps muscle is a unique occurrence in global literature.
In the reported case [13], significant functional limitations in elbow extension prompted total tumor resection, including that of a cuff of surrounding muscle. En bloc resection during surgery contributed to restoring normal elbow extension without the necessity for additional procedures. Early initiation of postoperative rehabilitation played a crucial role in the swift recovery. The case highlights the importance of surgical intervention in managing intramuscular hemangiomas, especially with functional impairment.
The unexpected coexistence of an abscess within the biceps brachii muscle adds a layer of complexity to this rare case. Abscesses in the biceps are uncommon, and their diagnosis can be challenging due to atypical presentations and the potential lack of classical signs associated with abscesses [3,5]. Predisposing conditions for biceps abscesses include immunocompromised state [4] or trauma [1]. As seen in the presented case, gastric cancer may be an underlying condition.
The presented case of intramuscular hemangioma with abscess formation in the biceps brachii muscle is a unique occurrence in the global literature. The discussion highlights the rarity of intramuscular hemangiomas, their management challenges, and the additional complexity introduced by the coexistence of an abscess in the biceps muscle. This case underscores the need for a thorough diagnostic approach, including imaging modalities like MRI, for accurate identification. The rarity of both intramuscular hemangiomas in the biceps brachii and their coexistence with abscess formation emphasizes the importance of continued research and shared clinical experiences to enhance understanding and optimize management strategies for such cases.
This case highlights the importance of considering soft tissue infections in patients, particularly in immunocompromised individuals. Despite the rarity of biceps muscle abscesses, healthcare providers should conduct thorough clinical examinations of extremities in applicable patients to ensure timely diagnosis and intervention. In conclusion, this case contributes valuable clinical insight into the diagnostic and management challenges associated with intramuscular hemangiomas and their rare coexistence with abscess formation in the biceps brachii muscle. Increased awareness and understanding of this uncommon clinical entity are crucial for improved future diagnosis and management. This case sheds light on the diagnostic challenges associated with intramuscular hemangiomas and their rare coexistence with abscess formation in the biceps brachii muscle. The complex clinical presentation emphasizes the need for a comprehensive diagnostic approach, including imaging modalities like MRI, for accurate identification. Surgical excision and early rehabilitation were instrumental in achieving optimal postoperative outcomes in this case. This contribution aims to enhance awareness and understanding of this uncommon clinical entity for improved future diagnosis and management.
PDF Links
PubReader
ePub Link
XML Download
Full text via DOI
Download Citation
Print
