Acral calcified angioleiomyoma

ARTICLE

Auteur(s) : Erika Sakai, Emiko Asai, Toshiyuki Yamamoto

Department of Dermatology and Plastic Surgery, Fukushima Medical University, Fukushima, 960-1295, Japan

Angioleiomyomas (vascular leiomyomas) are relatively common benign tumors, occurring more commonly among women. The tumors occur most frequently on the lower extremities, but rarely involve acral sites. Histologically, an angioleiomyoma is subdivided into the solid, cavernous and venous types. The solid type angioleiomyoma is composed of closely compacted smooth muscle and small, slit-like vascular channels. On the other hand, secondary calcification in angioleiomyoma is rare [1]. Recently, angioleiomyomas on acral sites which show extensive calcification have been reported, and the term “acral calcified vascular leiomyoma of the skin” was proposed for this rare, clinicopathological variant of angioleiomyoma [2]. Here we present an additional case.

A 75-year-old Japanese female consulted for a painful nodule on the sole, which had gradually increased in size since its appearance 25 years ago. The patient stated that there was no episode of antecedent trauma, nor any history of renal or endocrinal disease. A physical examination revealed a 2 × 2 cm-sized, firm, intradermal nodule with a smooth surface in the center of the right sole (figure 1A). Ultrasound scanning showed an oval hypoechoic structure containing echo-rich areas with dorsal echo-extinction (figure 1B), which suggested large areas of calcification within the mass. Histological examination of the totally excised nodule showed a circumscribed, partially encapsulated mass in the dermis (figure 1C). The tumor was composed of spindle to oval shaped smooth muscle cells with elongated nuclei, which were immunoreactive for α-smooth muscle actin (α-SMA), and slit-like vascular channels (figure 1D). Notably, extensive calcification consisting of variously-sized, irregular, amorphous basophilic masses was also prominent within the tumor (figure 1E).

Although dystrophic calcification in angioleiomyoma is described in textbooks, we encounter very few actual reports in the English literature. Meheshwari et al. [1] reported 4 cases of calcified angiomyoma of the foot. With the exception of one case, all the patients were over the age of 70. Two patients complained of pain, indicating that calcified angiomyomas should be considered in the differential diagnosis of painful nodules of the foot. Kacerovska et al. [2] recently presented 3 cases of angioleiomyoma with extensive calcification, and described the results of the histopathological examination in detail. Irregular, massive calcification was prominent within the tumor mass, as well as a number of small, circular, calcium deposits. All the patients were elderly females. The sites were in acral regions such as the index finger, sole and heel. Two patients complained of pain. Our case was also that of an elderly female, and involved painful lesions on the sole.

Dystrophic calcification has been reported to occur due to minor pressure, injury, previous inflammatory process, or de novo. Although the pathogenesis of diffuse calcification in angioleiomyomas is obscure, the tumors in the acral areas may be susceptible to calcification due to minor, repetitive injuries. Other factors such as degenerative changes and ischemia may also be involved.

Several, uncommon, histopathological features of angioleiomyomas have been reported to date, including epithelioid, palisaded, intravascular, and pleomorphic variations [3-6]. Prominent calcification should be considered an additional feature. In conclusion, angioleiomyoma with extensive calcification is a rare variant, and examination by ultrasonography before operation may serve as a readily available and useful tool for the prediction of extensive, diffuse calcification in angioleiomyomas.

Acknowledgements

References

2 Kacerovska D, Michal M, Kreuzberg B, Mukensnabl P, Kazakov DV. Acral calcified vascular leiomyoma of the skin: A rare clinicopathological variant of cutaneous vascular leiomyomas. J Am Acad Dermatol 2008; 59: 1000-4.

3 Heffernan MP, Smoller BR, Kohler S. Cutaneous epithelioid angioleiomyoma. Am J Dermatopathol 1998; 20: 213-7.

4 Baugh W, Quigley MM, Barrett TL. Palisaded angioleiomyoma. J Cutan Pathol 2000; 27: 526-8.

5 Sajben FP, Barnette DJ, Barrett TL. Intravascular angioleiomyoma. J Cutan Pathol 1999; 26: 165-7.

6 Kawagishi N, Kashiwagi T, Ibe M, et al. Pleomorphic angioleiomyoma. Am J Dermatopathol 2000; 22: 268-71.