Foot and ankle angioleiomyoma: a systematic review

Abstract

Background

Angioleiomyoma is a benign tumor arising from smooth muscle that is commonly found in the foot and ankle. This systematic review aims to synthesize the limited data from numerous case reports and case series, which often involve small sample sizes, to provide orthopaedic surgeons with a comprehensive overview of the diagnosis and management of foot and ankle angioleiomyoma.

Methods

This systematic review, performed following the PRISMA guidelines, brings updated information for the diagnosis and management of foot and ankle angioleiomyoma. 62 relevant studies were included. We analysed patient demographics, clinical characteristics, diagnostic workup, treatment, and clinical outcomes.

Results

Angioleiomyoma is more prevalent in middle-aged women, and pain is the most common symptom. Its diagnosis is often delayed due to its rarity and nonspecific presentation. Plain radiographs, MRI, and ultrasound of the foot and ankle are the most common preoperative imaging exams. Surgical excision is the treatment of choice with a low rate of both recurrence and malignant transformation.

Conclusions

This review emphasizes the importance of considering angioleiomyoma in the differential diagnosis of foot and ankle tumors and highlights the need for a comprehensive workup to improve diagnostic accuracy and ensure appropriate management.

Supplementary Information

The online version contains supplementary material available at 10.1186/s12891-025-08485-3.

Background

Angioleiomyoma is a benign tumor originating from the smooth muscle of the tunica media of the blood vessels, accounting for approximately 70 to 80% of mesenchymal tumor [1, 2]. It constitutes about 4.4% of all benign soft tissue neoplasms and accounts for 0.2% of tumors affecting the foot and ankle (F&A). Although angioleiomyomas can develop anywhere in the body, they are commonly found in the lower extremities [3–5]. Typically, these tumors occur most frequently in females aged between 30 and 50 years old [3, 6]. While factors like trauma, infection, hormonal changes, genetic influences, and vascular malformations are commonly associated with angioleiomyoma, its precise etiology remains uncertain [4, 7].

Angioleiomyoma typically manifests as a solitary and painful mass in the subcutaneous tissue. The differential diagnosis includes giant cell tumor, desmoid tumor, tophi, neuroma, schwannoma, sarcoma, ganglion cyst, dermoid cyst, epidermoid cyst lipoma, fibroma, glomus tumor and superficial acral fibromyxoma [6, 8]. The pain often stems from the compression of small interstitial nerve fibres and vessels within the tumor and resulting ischemia [4, 9, 10]. The physical and psychological implications of angioleiomyoma can significantly impact the quality of life for affected individuals.

This systematic review aims to provide orthopaedic surgeons with the most current and useful information regarding the diagnosis and management of F&A angioleiomyoma. Specifically, this review focuses on assessing and organizing the current preoperative work-up and analysing prognosis. By examining the pre-operative evaluation protocols reported in the literature, F&A surgeons can enhance their ability to recognize and treat this potentially hazardous tumor, ultimately improving symptom management and optimizing survival rates.

Methods

Information sources and search strategy

This systematic review was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines [11]. A comprehensive search was performed in electronic databases including MEDLINE via PubMed, and Google Scholar from inception to December 2024. The search strategy employed a combination of medical subject headings (MeSH) and keywords related to “angioleiomyoma” AND “foot” or “ankle”. The search was restricted to articles published in English, French, German, Spanish, and Portuguese. Two independent reviewers (MM and SS) screened the titles and abstracts of the identified articles to determine their eligibility for full-text review. Disagreements were resolved through discussion and consensus. The full texts of the selected articles were assessed for eligibility based on the inclusion and exclusion criteria. A third, senior author, was available to arbitrate but was not needed. The inclusion and exclusion criteria are summarized in Table 1.

Table 1.

Summary of inclusion and exclusion criteria

Inclusion criteria	Exclusion Criteria
All Level I - V published studies.	Reported location other than foot and ankle.
Studies written in English, French, German, Spanish or Portuguese.	Imprecise diagnosis (leiomyoma).
Study reports “foot” or “ankle” and “angioleiomyoma” or synonym (angiomyoma, vascular leiomyoma).	No details on which patients had which differential diagnosis, pre-operative exam, detailed treatment, etc.

Data collection

Data was extracted from the eligible studies using a standardized data extraction form, including study characteristics (e.g., author, year, study design), patient demographics, diagnostic methods, treatment modalities, outcomes, and follow-up duration (Table 2). A narrative synthesis of the findings was performed due to the heterogeneity of the included studies in terms of study design, interventions, and outcome measures.

Table 2.

Summary of each Article’s: primary author; year of publication; tumor location; age; sex; differential diagnosis; average time to surgery; work up; treatment; outcome and histology. F: female, M: male, S. E.: surgical excision

Author	Year	Location	Age (years)	Sex	Differential Diagnosis	Average time to surgery		Work up	Treatment	Outcome	Histology
Matsuda	2024	Forefoot	72	F	Hallux Valgus’s bursitis		Xray; MRI		S. E.		Angioleiomyoma
Kalia	2024	Forefoot	69	F	Schwannoma		US; MRI		S. E.		Angioleiomyoma
Kozlov	2024	Ankle	28	F	neoplasms		MRI		S.E.	2 years no recurrence	Angioleiomyoma Solid sub-type
El Jazouly	2024	Ankle	48	F			US		S. E		Angioleiomyoma
Gargouri	2023	Ankle	47	F	Schwannoma	2 years	Xray; US		S. E.	2 months no symptoms	Angioleiomyoma
Palacio Fernandez	2022	Forefoot	57	F		8 years	US		S.E.	3 months no symptoms	Angioleiomyoma
Inokuchi	2022	Forefoot	64	M			US; MRI		S: E.	1 year no recurrence	Angioleiomyoma
Lesbazeilles (3)	2022	Forefoot	66	M	Glomus tumor SAF	5 years	MRI		S. E.	-	Angioleiomyoma Superficial Axial Fibromyxoma
Irfan	2022	Midfoot	78	M	-	5 years	Xray; US; MRI		S. E.	-	Angiomyoma with calcifications
Inokuchi	2022	Forefoot	64	M	-	-	US; MRI		S. E.	12 month no recurrence	Angioleiomyoma
Suarez-Penaranda	2021	Midfoot Hindfoot Hindfoot	68 69 80	F M M	Epidermoid cyst	-	- - Xray		S. E. S. E. S. E.	- - -	Angioleiomyoma with calcifications Angioleiomyoma with calcifications Angioleiomyoma with calcifications
Ho	2021	Forefoot	80	M	Giant cell tumor Desmoid tumor Tophi Schwanomma	10 years	US; MRI; Biopsy		S. E.	12 months no recurrence	Angioleiomyoma
Bodapati	2021	Ankle Midfoot	34 50	M F	Neuroma Sarcoma	- 1 year	US US; MRI; Biopsy		S. E. S. E.	4 month no recurrence 10 weeks, no recurrence	Angioleiomyoma Angioleiomyoma
Fairbain	2021	Forefoot	55	F	-	-			S. E.	8 weeks no recurrence	Angioleiomyoma
Hsieh	2021	Midfoot Hindfoot Hindfoot	72 71 75	F F F	- - -	10 years 20 years 20 years	US US US		S. E. S. E. S. E.	- - -	Angioleiomyoma with calcifications Angioleiomyoma with calcifications Angioleiomyoma with calcifications
Edwards	2021	Forefoot	59	F	Neuroma	12 months	US		S. E.	12 months no recurrence	Angioleiomyoma
Rosell-Diaz	2019	Forefoot	68	F	-	7 months	Xray; US		S. E.	-	Angioleiomyoma
Taege	2019	Forefoot	8	F	Cyst	-	-		Incomplete excision	1 month no recurrence	Angioleiomyoma
Moriarty	2019	Hindfoot	45	M	-	6 years	MRI		S. E.	-	Angioleiomyoma; Mixed solid and venous sub-type
Ciaramella	2019	Hindfoot	55	F	Pseudoaneurysm of posterior tibial artery	3 months	Xray; US; MRI		S. E.	No symptoms, no recurrence	Angioleiomyoma
Avila Souza Junior	2019	Hindfoot	18	F			Xray; MRI		S. E.	Recurrence at 2 years	Angioleiomyoma
Sedberry	2018	Forefoot	39	F	Schwannoma	-	Xray; MRI		S. E.	12 months no recurrence	Angioleiomyoma
Lepoff	2018	Forefoot	68	M	-	3 years	US; MRI		S. E.	-	Angioleiomyoma with calcifications; Solid subtype
Alonzo Pena	2018	Midfoot	38	M	Fibroma Neurogenic tumor	-	US; MRI		S. E.	-	Angioleiomyoma
Sprinkle	2017	Hindfoot	64	M	-	5 years	XRay		S. E.	12 months no recurrence	Angioleiomyoma
Bartoli	2017	Ankle	41	M	-	-	Xray; US; MRI		S. E.	6 months no recurrence	Angioleiomyoma; Solid subtype
Nakale	2017	Ankle	67	F	-	3 months	US		S. E.	1 year no recurrence	Angioleiomyoma
Wollina	2017	Hindfoot	69	F	-	10 weeks	US		S. E.	-	Angioleiomyoma; Venous subtype
Baarini	2016	Midfoot	51	X	Fibroma Lipoma	2 years	Xray; US		S. E.	12 months no recurrence	Angioleiomyoma
Marco	2016	Hindfoot	68	F	-	7 years	Xray		S. E.	12 months no recurrence	Calcified angioleiomyoma
Thung	2016	Ankle	83	M	Pigmented villonodular synovitis	2 years	Xray; MRI		S. E.	-	Angioleiomyoma; Solid subtype
Sampaio	2015	Forefoot	53	M	-	4 years	-		S. E.	12 months no recurrence	Calcified angioleiomyoma
Blalock	2015	Midfoot	71	F	-	15 years	Xray		S. E.	-	Calcified angioleiomyoma
Oiso	2013	Midfoot	65	M	Plantar epidermoid cyst (HPV infection)	1 year	US; Biopsy		S. E.	-	Angioleiomyoma
Gajanthodi	2013	Forefoot	44	M	Lipoma Dermoid cyst Ganglion	3 months	-		S. E.	-	Angioleiomyoma
Lemtibbet	2013	Midfoot	66	M	Angiolipoma Liposarcoma	6 years	US		S. E.	-	Calcified angioleiomyoma; Solid sub-type
Cheung	2012	Hindfoot	50	F	-	3 years	Xray; MRI		S. E.	3 years no recurrence	Angioleiomyoma
Hamoui	2010	Ankle	64	F	-	-	Xray; MRI; EMG		S. E.	12 months no recurrence	Angioleiomyoma
Sakai	2010	Midfoot	75	F	-	25 years	US		S. E.	-	Calcified angioleiomyoma
Gomez-Bernal	2010	Hindfoot	52	F	-	3 years	-		S. E.	-	Calcified angioleiomyoma; Solid sub-type
Welborn	2010	Ankle	63	F	-	5 years	MRI		S. E.	-	Angioleiomyoma
Kadowaki	2010	Hindfoot	77	F	Calcified aneurysm	1 year	Xray; CT; MRI		S. E.	2 years no recurrence	Angioleiomyoma; Solid sub-type
Maheshwari	2008	Midfoot Forefoot Forefoot Forefoot	35 75 73 73	M F M M	- - - -	3 months 30 years 6 years 6 months	Xray; MRI Xray; CT; MRI Xray Xray; MRI		S. E. S. E. S. E. S. E.	12 years no recurrence 9 years no recurrence 10 years no recurrence 10 years no recurrence	Calcified angioleiomyoma Calcified angioleiomyoma Calcified angioleiomyoma Calcified angioleiomyoma
Kacerovska	2008	Midfoot Hindfoot	72 57	F F	- -	5 years 1 year	- -		S. E. S. E.	- -	Calcified angioleiomyoma Calcified angioleiomyoma
Gupte	2008	Midfoot Foot Foot	54 69 40	F F F	Soft tissue tumor Synovial sarcoma Sarcoma	2 years - 2 years	MRI Xray; MRI MRI		S. E S. E. S. E.	2–32 months no recurrence	Angioleiomyoma; Solid sub-type Angioleiomyoma; Solid sub-type Angioleiomyoma with hemorrhage
Murata	2007	Hindfoot Hindfoot	58 63	F F	- -	5 years 1 year	Xray; MRI; Scintigraphy Xray; MRI; Scintigraphy		S. E. S. E.	3 years no recurrence 18 months no recurrence	Calcified angioleiomyoma; Solid sub-type Calcified angioleiomyoma; Solid sub-type
Cancilleri	2007	Ankle	48	F	Tarsal tunnel syndrome related to a metabolic disease	1 year	EMG; MRI		S. E.	12 months no recurrence	Angioleiomyoma
Sonohata	2007	Hindfoot	47	M	-	2 years	Xray		S. E.	7 months no recurrence	Angioleiomyoma
Ramesh	2004	Midfoot	51	F	Dermoid implantation	18 months	-		S. E.	-	Angioleiomyoma
Yates	2001	Forefoot Forefoot Forefoot	78 49 41	F M F	Ganglion - -	12 years 14 years 4 years	- Xray Xray		S. E. S. E. S. E.	12 months no recurrence 12 months no recurrence 12 months no recurrence	Calcified angioleiomyoma; Venous sub-type Angioleiomyoma; Solid sub-type Angioleiomyoma; mixed solid and venous sub-type
Santucci	2000	Midfoot	52	F	Benign cyst	1 year	Xray		S. E.	4 years no recurrence	Angioleiomyoma
Pastore	1999	Hindfoot	38	M	Ganglionic cyst	2 months	Xray		S. E.	Recurrence at 2 months	Angioleiomyoma
Kinoshita	1997	Midfoot,	61	F	-	-	MRI			15 months, no recurrence	Angioleiomyoma; Solid sub-type
Hanft	1997	Ankle Midfoot	48 47	F F	- -	3 years 10 years	Xray; Aspiration Xray; Aspiration		S. E. S. E.	- -	Calcified Angioleiomyoma ; Venous sub-type Angioleiomyoma ; Venous sub-type
Habershaw	1994	Forefoot	48	M	-	10 years	Xray; MRI		S. E.	-	Calcified angioleiomyoma
Requena	1993	Forefoot	65	F	-	-	Xray		S. E.	-	Angioleiomyoma
Craigen	1991	Forefoot Forefoot	40 79	F F	Neuroma -	6 years 15 years	- -		S. E. S. E.	Recurrence after 5 years 3 years	Angioleiomyoma Angioleiomyoma
Sawada	1988	Forefoot	53	F	Ganglion	2 years	-		S. E.	-	Angioleiomyoma with significant mixoid degeneration
Genakos	1987	Ankle	57	M	-	10 years	-		S. E.	-	Angioleiomyoma
Brenner	1986	Forefoot	74	F	-	10 years	Xray		S. E.	-	Calcified angioleiomyoma
Sweeney	1983	Forefoot	28	M	-	10 years	Xray		S. E.	-	Angioleiomyoma
Stout	1937	Ankle Ankle	42 41	F F	Fibroma Ganglion	2 years 4 years	- -		S. E. S. E.	- -	Calcified angioleiomyoma Angioleiomyoma

Risk of bias

Two authors reviewed all the studies (MM and SS). No automatic tool was used.

Statistical analysis

Descriptive statistics were employed to summarize the demographic characteristics of the study population, encompassing age, gender distribution, and the anatomical sites affected by angioleiomyoma. All data were collected in a database (Excel for Windows, version 6.0.14026.20202, Microsoft Cooperation, USA). Statistical analyses were performed using IBM^® SPSS Statistics for Windows^®, version 23 (IBM Corp., Armony, N.Y., USA). Qualitative variables were analyzed using frequencies, percentages, and standard deviations (SD), while quantitative variables were assessed using means and ranges.

Results

Studies

A total of 1,529 studies were identified during the initial search. After reviewing the titles and abstracts, articles that were repeated across different sources or written in languages other than English, French, Portuguese, German, or Spanish were excluded. This resulted in 98 studies being selected for abstract or full-text review. Of these, 36 studies were excluded for various reasons, including lesions located outside the foot and ankle region, imprecise diagnoses such as leiomyoma, or incomplete patient data related to differential diagnoses, pre-operative examinations, or surgical procedures.

Among these, 62 studies met the inclusion criteria for further analysis (Fig. 1). No meta-analyses or systematic reviews were found in the literature. The year of publication for these studies ranged from 1937 to 2024.

Fig. 1.

PRISMA flow diagram illustrating the study selection process. The diagram outlines the progression from the initial literature search (1,529 studies) to the final inclusion of 62 studies. Key exclusion criteria are detailed, including language restrictions, anatomical location (foot and ankle), diagnosis specificity, and completeness of patient data regarding differential diagnoses, pre-operative evaluations, and interventions

Patients

A total of 79 patients with angioleiomyomas were described in the 62 included studies. The mean age of the patients was 57.3 ± 3.4 years, ranging from 8 to 80 years old. The male-to-female ratio was 1:1.5, with 64.6% of the patients being female. In 78 cases out of 79 (98.6%), the pre-operative symptomatology was described. 51 patients (64.6%) complained of pain, 20 patients (25%) of difficulty with footwear, 13 patients (16.5%) of a swelling or mass and 10 (12.7%) of problems with walking.

Location

For lesion location, 14 out of 77 cases (18.2%) were located at the ankle, 18 cases (23.4%) at the hindfoot, 17 cases (22.1%) at the midfoot, and 28 cases (36.4%) at the forefoot. Two cases (2.9%) were reported as “foot” without further specification.

Clinical reasoning

The differential diagnosis for angioleiomyoma included pathologies such as giant cell tumor, desmoid tumor, tophi, neuroma, schwannoma, sarcoma, ganglion cyst, dermoid cyst, epidermoid cyst, lipoma, fibroma, glomus tumor, and superficial acral fibromyxoma. Only 40.5% of the studies (32 out of 79 cases) reported a list of differential diagnoses prior to performing imaging. None of the studies reported angioleiomyoma as the primary diagnosis, or included it in the list of differential diagnoses, prior to surgery.

Pre-operative work-up for diagnosis

The pre-operative diagnostic work-up involved plain radiographs of the foot and ankle as the primary examination in 35 out of 79 cases. Among these 35, only 20 patients underwent further examinations. For the other 15, surgeons found X-ray sufficient to proceed to surgical excision. Magnetic resonance imaging (MRI) was the second most used modality in 40.5% (32/79) of cases. MRI showed a hyper-intense signal of the lesion on T2 sequence in 59.4% (19/32) of instances. There was no other specific sign for the other lesions. Ultrasound (US) was realized in 31.6% (25/79) of cases. Biopsy was performed in only 3 patients (3.8%) before surgical intervention. Two patients (2.5%) underwent scintigraphy, and 3 patients (3.8%) underwent electromyography (EMG) prior to surgery. Approximately one-fifth of the patients (19%) did not undergo any preoperative investigation. The results are summarized in Table 2.

Surgical treatment

Surgical excision was the treatment of choice for all patients, with an average time to surgery of 5.6 ± 1.6 years from the onset of symptoms. A total of 78 out of 79 patients underwent an in toto surgical excision [12]. The outcomes were described in 44 out of 79 cases. Some authors reported a period without recurrence of symptoms ranging from 1 month to 12 years after surgery, with a mean follow-up time of 26.7 ± 11.7 months. Of the 79 cases, 25 (31.6%) included descriptions of the post-operative symptoms. They were described as asymptomatic or pain free during follow-up. Only one patient reported a tender scar, which improved during follow-up and was no longer present at the last consultation. Recurrence was observed in two patients (2.8%), one after 2 years and the other after 5 years [13, 14]. Only one underwent a second surgery, resulting in complete relief of symptoms and no further recurrence. The other refused a second surgery, as symptoms were minimal.

Histology

Histologic examination was performed on all excised tumors, and calcifications were noted in 22 angioleiomyomas (27.8%). Among the 18 cases that reported subtype classification, 12 (66.7%) were classified as solid subtype, 4 (22.2%) as venous type, and 2 (11.1%) as mixed type (Table 2).

Discussion

Epidemiology

Angioleiomyoma is more commonly found in women, which is consistent with the findings of Hachisuga et al., who reported the same male-to-female ratio found in the 62 reviewed studies (1:1.5) [3]. In the literature, angioleiomyoma is typically described as a painful tumor occurring between the 3rd and 6th decades of life [3, 6]. In this review, the patients’ mean age at the time of surgery was 57.3 ± 3.4 years. There was no site predilection for F&A angioleiomyomas, as they were almost equally distributed among the ankle, hindfoot, midfoot, and forefoot.

Diagnosis

Due to the rarity of angioleiomyoma, it is often not considered as a possible in the list of differential diagnoses prior to imaging or even surgical excision. Clinical diagnosis is challenging, as there are no specific signs or symptoms associated with angioleiomyoma. The presentation of angioleiomyoma is like many other F&A tumors and pain is not specific to angioleiomyoma alone. This might explain the relatively long mean time to surgery (5.6 ± 1.6 years) that we observed in these studies. Currently, there is no specific gold-standard imaging technique for the diagnosis of these vascular tumors. The diagnostic workup varied significantly among the reviewed studies, with MRI, X-ray, ultrasound, aspiration, scintigraphy, CT, and EMG with nerve conduction being among the diagnostic tools used (Table 2). In some cases, the diagnosis is only mentioned after surgery and pathologic assessment, leading to a lack of pre-operative workup. Angioleiomyoma is rarely diagnosed before surgery, contributing to delays in surgical intervention.

F&A radiography may reveal calcifications in some angioleiomyomas (27.8% of all foot and ankle cases). These are rare in acral locations, accounting for 2.0–3.3% of cases [3, 4, 15]. The high percentage of calcified angioleiomyomas found in F&A cases suggests either a location-dependent difference or an underestimation in the literature [16, 17]. Furthermore, authors suggest that minor and repetitive trauma in peripheral tumoral areas may contribute to the development of calcifications [18, 19].

On ultrasound, angioleiomyomas typically present as subcutaneous, solid, and homogeneous tumors with smooth or lobulated margins [20]. Small vessels with blood flow are often observed within an echogenic background, and a feeding vessel can often be identified. However, some tumors did not show blood flow on Doppler, likely due to tumor characteristics such as calcification or small size (< 10 mm). Calcifications, ranged from minor deposits to complete calcification, are considered rare in angioleiomyomas, potentially leading to misdiagnosis [21]. A lesion can be accurately diagnosed as an angioleiomyoma using ultrasound if all characteristic signs—such as a subcutaneous, solid, and homogeneous tumor with smooth or lobulated margins, the presence of small vessels with blood flow, and a feeding vessel—are present.

MRI reveals well-defined, round, or oval tumors, mostly bordered by a hypointense lining corresponding to a fibrous capsule [22]. Smooth muscle and numerous vessels within the tumor result in a hyperintense signal on T2-weighted MRIs [23]. Marked gadolinium enhancement can be observed in more vascular portions of the lesions [24]. The tumoral tissue is slightly hyperintense compared to muscle on T1-weighted images [23]. The “dark reticular sign”, which is described as hypo- or iso- intense linear and/or branching structures on T2 weighted images, has been defined as characteristic finding of angioleiomyoma. However, MRI cannot differentiate between all three subtypes of angioleiomyoma. A lesion can be diagnosed as an angioleiomyoma on MRI if characteristic features are present, including a well-defined, round or oval shape with a hypointense fibrous capsule, hyperintense signals on T2-weighted images due to smooth muscle and vascularity, and the presence of the ‘dark reticular sign,’ which is considered a hallmark finding of this tumor.

Although certain signs on ultrasound or MRI may suggest a specific subtype, histological examination is still required for accurate classification [22, 25, 26].

Surgical excision

In toto surgical excision is necessary for several reasons. First, it aims to relieve the pain experienced by symptomatic patients. Second, in the absence of a clear diagnosis, complete removal (excisional biopsy) of the tumor is the most appropriate procedure. Finally, surgical excision helps prevent recurrence and minimizes the risk of malignant transformation, as this risk is rare, but accounts for 7% of soft tissue sarcoma [27]. As demonstrated in this review, the outcome after surgical excision is excellent, with a low recurrence rate observed in 2 out of 79 patients.

Outcome

44 studies provided information on the outcome after surgery, with a wide range of follow-up times (1 month to 12 years). The average follow-up time was 26.7 ± 11.7 months. Recurrent tumors were reported in two cases (one after 2 years and the other after 5 years), but no malignant transformation was observed [13, 14]. One patient achieved symptom relief following the original surgical excision, while the other patient declined a second surgery, making it impossible to exclude the possibility of malignant transformation. The review indicated that when an in toto surgical excision is performed, recurrence of symptoms or the disease is unlikely, resulting in minimal likelihood of malignant transformation.

Histology

Angioleiomyoma is now recognized as an independent tumor entity by the World Health Organization, forming a morphological continuum with myopericitoma and myofibroma [28]. Histologically, angioleiomyoma exhibits two-layer circumferentially arranged vascular channels [29]. Bundles of mature smooth muscles are oriented around the blood vessels.

While some authors consider angioleiomyoma to be a vascular malformation derived from arteriovenous anastomoses, similar to glomus tumors, others suggest a hamartomatous origin [3–5, 30]. Duhig et al. proposed that these lesions develop sequentially from smooth muscle proliferation within a vascular hamartoma, leading to the formation of an angiomyoma [4]. Eventually, continuous smooth muscle proliferation results in a simple leiomyoma.

Angioleiomyomas are classified into three histopathological subtypes [31]:

• Solid subtype: characterized by small slit-like vascular channels, accounting for 66% of all angioleiomyomas.

• Venous subtype: displaying vascular channels with thick muscular walls that are distinct from the surrounding intervascular smooth muscle, accounting for 23% of cases.

• Cavernous subtype: characterized by dilated vascular channels in which the blood vessel wall blends imperceptibly with the smooth muscle proliferation, accounting for 11% of cases.

Although a classification into three subtypes exists, only 18 cases in the review were classified as solid, venous, or cavernous subtypes [31]. The prognosis and treatment do not depend on the specific subtypes. Since it is mainly a histological classification without clinical relevance, it may not be mentioned in every study.

Angioleiomyoma’s malignant progression into angioleiomyosarcoma has been reported in 1% of cases [27, 32]. This is a rare event, and angioleiomyosarcoma represents 7% of all soft tissue sarcomas [33]. Foot and ankle angioleiomyomas, typically associated with significant pain, may result in shorter time to surgery, reducing the likelihood of malignant transformation.

Strengths and limitations

This study has several limitations that should be acknowledged. First, the search strategy was limited to two databases, MEDLINE via PubMed and Google Scholar, instead of including broader sources such as Scopus. This choice was influenced by accessibility and the practical focus on platforms commonly used by clinicians. While it is possible that some studies indexed in other databases may not have been included, a significant portion of the relevant literature is represented in our review. Given the nature of the existing studies, they are likely small in scale with minimal impact on the overall statistical analyses. Additionally, the review relies heavily on case reports and small case series, given the rarity of foot and ankle angioleiomyoma. This limited the sample size and may have introduced publication bias, as rare or atypical cases are more likely to be reported. Furthermore, the study relies on historical data from older case reports and series, which may lack the level of detail and methodological rigor seen in more recent studies. This reliance on older data could introduce bias and limit the generalizability of our findings. The reliance on a small number of databases and cases highlights the need for more comprehensive research and collaborative efforts to better understand this rare tumor.

Bernard et al. [34] reported a case series of 142 patients, describing the clinical characteristics, radiological features, histopathological findings, and treatment outcomes of angioleiomyomas. They performed cross-referencing between clinical data, MRI results, and histopathological findings but did not find any statistically significant results. They concluded that, although MRI can be beneficial as a diagnostic instrument, final confirmation by histopathological analysis is mandatory. Unfortunately, this study could not be fully incorporated into our review, as it lacked clarity regarding the association between specific patients and their respective diagnostic evaluations or treatments.

Despite these limitations, this systematic review represents a significant strength as the only comprehensive review specifically addressing foot and ankle angioleiomyoma. It provides detailed and complete information on the diagnosis, clinical presentation, and imaging findings of this rare tumor. Moreover, it offers highly relevant and practical insights into treatment options, assisting clinicians in improving patient outcomes. By synthesizing available data, the review bridges critical gaps in the current understanding of this condition.

Conclusion

Angioleiomyoma should be considered in the differential diagnosis of any soft tissue mass in the foot and ankle, especially in middle-aged women presenting with painful lesions. Both ultrasound and MRI play a crucial role in the preoperative evaluation of these tumors. If the classic characteristics are identified—such as the “dark reticular sign” on MRI or vascular flow and feeding vessels on ultrasound—the diagnosis can often be suggested prior to surgery. These imaging modalities provide valuable insights that aid in planning appropriate treatment.

Surgical in toto excision is the recommended treatment approach for angioleiomyoma, aiming to relieve symptoms, achieve a definitive diagnosis, and prevent recurrence. Neglecting angioleiomyoma can lead to delays in treatment or inappropriate treatment, increasing morbidity and the potential risk of malignant transformation.

Further research is needed to enhance our understanding of the aetiology, pathogenesis, and optimal management strategies for foot and ankle angioleiomyoma.

Electronic Supplementary Material

Below is the link to the electronic supplementary material.

Author contributions

In the course of this study, M.J.M. and S.S. have made substantial contributions across multiple domains, including the conceptualization and design of the research, the acquisition, analysis, and interpretation of data, as well as the drafting and critical review of the manuscript to ensure its intellectual integrity. Their commitment to being accountable for all aspects of the work and their willingness to review the final version prior to publication underscore their dedication to scholarly rigor and excellence. Similarly, J.M.S and M.T. have demonstrated their commitment to upholding accountability and ensuring the quality of the final publication through their agreement to review the final version. A.S. in addition to agreeing to be accountable for all aspects of the work and reviewing the final version, has also provided substantial contributions to the conceptualization and design phase of the study, enriching its theoretical foundation and methodological approach.

Funding

The authors have no financial or proprietary interests in any material discussed in this article. We acknowledge that no funding was received for this research.

Data availability

The dataset supporting the conclusions drawn in this article is presented within the manuscript and summarized comprehensively in Table 2. All data necessary for interpretation, replication, and further analysis are included within the articles listed in Table 2. We have ensured that relevant information from the dataset is clearly outlined in Table 2, providing readers with access to the key findings and supporting details. Should there be any specific queries or requests for additional information regarding the dataset, we are prepared to address them promptly.

Declarations

Ethics of approval and consent to participate

As this manuscript presents a review study, it exclusively synthesizes and analyzes data already published in existing literature. Consequently, the nature of our research does not involve any direct involvement with human subjects or experimental procedures. Given that our study solely relies on previously published material and does not entail new data collection, ethical approval from an ethics committee or Internal Review Board (IRB) was not sought nor required.

Consent for publication

Not applicable.

Competing interests

The authors declare that they have no competing interests.