Pedal Angioleiomyoma In A Patient With CRPS: An Uncommon Neoplasm

Angioleiomyomas are benign soft tissue tumors arising from the smooth muscle layer, or tunica media, of blood vessels. These rare tumors present as painful nodules in the face and extremities, most commonly affecting women between 30 and 50 years old. Although this uncommon subcutaneous and encapsulated soft tissue mass most often occurs in the extremities, there are several documented cases found in the viscera; such as the uterus, bladder, lung, and gastrointestinal tract.^1,3

The exact etiology of this tumor is unknown, but some possible causative factors include infections, hormonal imbalance, and acute trauma. The leading sign of a symptomatic angioleiomyoma is paroxysmal pain, commonly initiated by light touch or environmental triggers. It is challenging to make a preoperative diagnosis of angioleiomyoma because it has no distinct findings, and magnetic resonance image (MRI) is useful, but not specific. Therefore, the tumor is rarely diagnosed until after excision and histopathological examination, making it difficult to diagnose clinically. The overall incidence of angioleiomyomas is unknown, as many are asymptomatic, and many benign lesions remain undetected. This case study reports an unusual occurrence of an angioleiomyoma at the central plantar midfoot.^2,3

When a Patient With Chronic Pain Presents With a Growing Mass

A 44-year-old female with CRPS (chronic regional pain syndrome) presented to the podiatry clinic with a painful soft tissue mass in the right plantar foot. She related that the mass, present for about two years, was increasing in size and causing pain secondary to fullness in the region. This was particularly uncomfortable when walking in shoes. The patient’s past medical history included chronic migraines and endometriosis. Current medications included tramadol and methocarbamol. She had experienced symptoms of CRPS for the past two years after a finger injury and three-level cervical fusion. Past surgical history includes a cervical fusion of C5, C6, and C7, and ovary removal. Her family history was noncontributory, the patient denied any tobacco use, but acknowledged social alcohol consumption.

Relevant review of systems included numbness and diffuse joint pain, with all other systems within normal limits. Focused physical exam included pedal pulses 2/4, normal capillary refill time, and no erythema or edema over affected area. Neurologically, there was intact epicritic and vibratory sensation, with tingling, sharp and shooting pain during palpation of the medial plantar nerve at the porta pedis on the right lower extremity. Dermatologically, there was a palpable nodular mass noted at the plantar medial aspect of her right arch, sitting just under the porta pedis, approximately 2.5 x 3.5 cm. With palpation, it caused some radiating discomfort proximally and distally.

Radiographic images of the right foot showed no osseous pathology. A multiplanar MRI without contrast (field of view centered about the mid- to forefoot) showed a rounded cystic lesion closely abutting the plantar aspect of the flexor hallucis longus tendon, appearing well-encapsulated, with thin internal septations noted. Small amounts of adjacent soft tissue edema surrounded the mass. The flexor hallucis longus tendon remained intact.

Pertinent Points in the Management and Outcome

The patient elected to have the mass removed after a full, detailed discussion of conservative and surgical options. Surgically, we made a curvilinear incision along resting skin tension lines, exposing the plantar fat pad. Blunt and sharp dissection deepened the incision to the plantar aspect of the flexor hallucis longus tendon, and allowed visualization of the mass. Once fully exposed, we excised the tumor, which was consistent with the MRI report. It was encapsulated, measured 3.3 x 1.8 x 1.0 cm, and presented with central hyaline degeneration and infarction. After irrigation, we performed the remaining soft tissue closure in layers, with a small drain placed to prevent possible hematoma formation. The postoperative course involved four-to-six weeks of guarded weight-bearing in surgical shoe, with limitations on driving.

The patient presented for follow-up visits and suture removal. At three weeks postop, she relates significant improvement. Despite CRPS, patient states that she experienced 5/10 pain, decreased from prior to surgery, and requested 10 mg hydrocodone for a few days. She related that gabapentin helped immediately, but that it causes her to stutter. Objectively, the patient was no longer in distress in regards to her foot, and had mild paresthesias dorsally, plantarly, and distally about surgical site. There was notable improvement with regard to palpable tenderness. Edema and erythema surrounding the surgical incision sites also markedly improved.

According to the literature, simple excision of angioleiomyoma is the definitive treatment,² and in this patient, there is no reoccurrence of this tumor to date. As for concluding thoughts, in this particular case, excision of the angioleiomyoma aided in reducing the effects of her chronic pain she suffers from CRPS.

Dr. Joshua is a third-year resident at Hunt Regional Medical Center, Greenville, TX.

Dr. Villaruz is a third-year resident at Hunt Regional Medical Center, Greenville, TX.

Dr. Brook is a Fellow of the American College of Foot and Ankle Surgeons and practices in Dallas, TX.

Dr. Brancheau is a Fellow of the American College of Foot and Ankle Surgeons and is the Podiatric Residency Director at Hunt Regional Medical Center in Greenville, TX.

1. Baarini O. Angioleiomyoma of the plantar-medial arch: a case report. J Clin Diagnost Res. 2016;10(7):PD07-PD08. doi:10.7860jcdr201619988.8112

2. Jalgaonkar A, Dachepalli S, Farid M, Rao S. (2011). Angioleiomyoma of the Knee: Case Series and an Unusual Cause of Knee Pain. J Knee Surg. 24(01), 033-038. doi:10.1055s-0031-1275403

3. Lepoff A, Makarov V, Williams M. (2018). Angioleiomyoma of the Plantar Foot. J Am Podiatr Med Assoc. 2018;108(3):262-266. doi:10.754717-071