ISSN 0972-978X 

 
 
 
 
 
 
 
 
 
 
 
 
  About COAA
 

 

 

 

 

 

 

CASE REPORT

Digital Angioleiomyoma: A Case Report and Review of the Literature

 Michael J. Sileo *

* Sports Fellow,Department of Orthopaedic Surgery,Lenox Hill Hospital

Address for Correspondence:  

Michael J. Sileo
Sports Fellow,Department of Orthopaedic Surgery,Lenox Hill Hospital,100 East 77th Street, New York

Abstract:

Angioleiomyomas are rare, benign, smooth muscle tumors that are rarely found in the hand.  In Butlerís review of 437 hand tumors, only one was a leiomyoma4.  We present a case of a digital angioleiomyoma that presented with gradual enlargement and reactive bony changes of the middle phalynx.  Wide excision proved to be curative.  A discussion and review of the literature follows. 

J.Orthopaedics 2008;5(2)e2

 Case Report:

A 60 year-old right-hand dominant attorney presented with a mass on the right index finger which had been present for several years.  The patient noted that it arose insidiously and reported no history of trauma.  He sought treatment as he felt that it had recently increased in size.  He denied any pain or recent weight loss, reported no history of cold intolerance, and was working at the time of presentation.

On physical exam, there was noted to be a firm mass approximately 1.5 cm in size located on the radial volar aspect of the distal portion of the index middle phalynx.  No skin changes were appreciated.  The mass was slightly mobile and minimally tender.  Sensation was normal on the radial and ulnar aspect of the index finger, and there was full function and range of motion of the flexor and extensor tendons to the digit.  Soft tissue swelling as well as some reactive changes of the middle phalynx of the index finger was appreciated on radiologic exam (Figure 1)

Differential diagnosis at the time of examination included giant cell tumor of tendon sheath, glomus tumor, ganglion, mucous cyst, and lipoma.

The patient chose to have the mass excised on an elective basis due to its recent increase in size.  The mass was excised under local anesthesia with sedation.  The mass was grossly firm and grayish in color.  It did not appear to involve the radial digital artery, vein, or nerve.  Pathologic analysis identified the mass as a completely excised angioleiomyoma approximately 0.9x0.9x0.7 cm in size.  It stained positively for smooth muscle actin and negative for immunostain S-100, supporting the diagnosis of angioleiomyoma (Figure 2).

Fig. 1:Soft tissue mass and apparent sclerotic changes in the radial and distal aspect of middle phalynx

Figure 2: Microscopic and immunohistochemical features of angioleiomyoma, solid type.  A. Hematoxylin and eosin, 40X amplification. B. Hematoxylin and eosin, 100X amplification. C. Hematoxylin and eosin, 400X amplification. D. Immunohistochemical stain for smooth muscle actin (200X amplification, clone 1A4, Ventana, Tucson, AZ).

Microscopic description: The lesion is a well demarcated nodule of smooth muscle tissue punctuated with thick-walled vessels (Figure 2 A, B) with small partially patent lumens (Figure 2 C).  It is immunoreactive for smooth muscle antigen (Figure 2 D)


Discussion:

Leiomyomas are benign soft tissue tumors that are distributed wherever smooth muscle is present1.  Angioleiomyomas, a subtype of leiomyomas, are rare, benign, smooth muscle tumors that arise from the tunical media layer of small arteries and veins2.  Angioleiomyomas may occur in the dermis, subcutaneous fat, and fascia3.

While these tumors may be found anywhere in the body, only rarely have they been described in the hand.  In 1937, Butler reviewed 437 hand tumors, only one of which was a leiomyoma4.  Duhig and Ayerís review of 60 angioleiomyomas revealed only 3 cases in the hand5.  In a review 85 upper extremity angioleiomyomas at their institution, Nevaiser et al noted that 12 involved the hand6.  In Hachisugaís comprehensive review of 562 cases, 56 were found to involve the hand2. 

Histologically, angioleiomyomas are divided into three subtypes: capillary or solid (most common), venous, and cavernous (least common)2,3,7.  The most common solid type has compacted smooth muscle and many small, slitlike vascular channels.  Thick muscular walls distinguish the venous type.  In the least common cavernous type, the vascular channels are dilated with less smooth muscle present2.  Attempts to definitively correlate histologic subtype with clinical presentation has proven inconclusive2,8.  Some have suggested that angioleiomyomas may in fact not be a tumor at all, but a vascular malformation5. A recently published case report of excision of this tumor from a digital artery with end-to-end repair of the artery by Calle et al may support this assertion1.   

It is estimated that approximately 50 - 60% of patients present with a chief complaint of pain related to the mass frequently associated with trauma or extreme cold9.  This may easily be confused with a glomus tumor, of which two-thirds are typically associated with pain, frequently intensified by cold exposure10.  Pain is a less common presenting complaint with upper extremity angioleiomyomas9.  Many authors have found pain to be associated with the solid subtype2,3,8.   Some have postulated that pain is due to local ischemia of the mass2.  Recently, however, nerve fibers have been visualized within these tumors at the microscopic level11.  

Angioleiomyomas typically occur in the fourth through sixth decades2,6.  They may be anywhere from 0.5-3 cm in size, most commonly being less than 1 cm3,6.  While these tumors can be found throughout the body, approximately 89% are reported in the extremities, most commonly below the knee in the lower extremity2,8.  Overall angioleiomyomas are more common in females compared to males (2.2:1)3, but there is a higher incidence of upper extremity angioleiomyomas in males11.  While they are more frequently found on the extensor surface of the lower extremity, these lesions appear to have a predilection for the volar surface of the hand11. Occasionally, these tumors may cause local bony changes12, as was found in this case.  This is likely due to local pressure effects.  Malignant degeneration is extremely uncommon, with only two reported cases found in the literature6,13.  In one case, ray resection was necessary for local control13.  Classically, local excision of the mass is the definitive treatment of choice.             

Summary :

Angioleiomyomas are rare, benign smooth muscle tumors that can occur anywhere in the body.  They are more common in the lower extremities, and are rarely found in the hand.  Pain is frequently present.  Malignant degeneration is extremely rare, and simple excision is typically curative.  Angioleiomyomas should be considered in the differential diagnosis of nodular masses of the hand.

References :

  1. Calle SC, Eaton RG, Littler JW.  Vascular Leiomyomas in the Hand.  J Hand Surg  1994 Mar;19(2):281-286 
  2. Hachisuga T, Hashimoto H, Enjoji M.  Angioleiomyoma: A clinico-pathologic reappraisal of 562 cases.  Cancer 1984;54:126-130 
  3. Freedman AM, Meland NB.  Angioleiomyomas of the Extremities: Report of a Case and Review of the Mayo Clinic Experience.  Plast Reconstr Surg. 1989 Feb;83(2):328-331 
  4. Butler ED, Hamill JP, Seipel RS, de Lorimier AA.  Tumors of the Hand: A ten-year survery and report of 437 cases.  Am J Surg  1960;100:293 
  5. Duhig JT, Ayer JP.  Vascular leiomyoma: A study of sixty cases.  Arch Pathol Lab Med 1959; 68:424-430 
  6. Neviaser RJ, Newman W.  Dermal Angiomyoma of the Upper Extremity.  J Hand Surg 1977;2:271-274 
  7. Dominguez-Cherit J, Brandariz A.  Distal Digital Angioleiomyoma: A Case Report and Review of the Literature.  Int J Dermatology 2004 Feb;42(2):141-143 
  8. Morimoto N.  Angiomyoma (Vascular leiomyoma): A clinicopathologic study.  Medical Journal of Kagoshima University 1973;24:663-683 
  9. Ramesh P, Annapureddy SR, Khan F, Sutaria PD.  Angioleiomyoma: A Clinical, Pathological and Radiological Review.  Int J Clin Pract  2004 Jun;58(6):587-591 
  10. Ozdemir O, Coskunol E, Ozalp T, Ozaksar K.  Glomus tumors of the finger: a report on 60 cases.  Acta Orthop Traumatol Turc. 2003;37(3):244-248 
  11. Lawson GM, Salter DM, Hooper G.  Angioleiomyomas of the Hand.  A Report of 14 Cases.  J Hand Surg (Br) 1995 Aug;20(4):479-83 
  12. Glowack KA, Weiss AP.  Vascular Leiomyoma of the Finger Causing Bone Erosion.  J Hand Surg 1995;20(6):1011-1013 
  13. Herren DB, Zimmermann A, Buchler U.  Vascular Leiomyoma in an Index Finger Undergoing Malignant Transformation.  J Hand Surg (Br) 1995;20B(4):484-487 
  14. Albares,MP, Belinchon I, Vergara G, Pascual JC, Pastor E.  Digital Angioleiomyoma.  Int J Dermatology 2002 Aug; 41(8):527 
  15. Oktem F.  Vascular Leiomyoma of the Hand.  Plast Reconstr Surg  2005 April;115(4):1218-1219 
  16. Stout AP. Solitary cutaneous and subcutaneous leiomyoma.  Am J Cancer  1937;29:435-69

 

This is a peer reviewed paper 

Please cite as : Michael J. Sileo : Digital Angioleiomyoma: A Case Report and Review of the Literature

J.Orthopaedics 2008;5(2)e2

URL: http://www.jortho.org/2008/5/2/e2

ANNOUNCEMENTS

 


 

Arthrocon 2011


Refresher Course in Hip Arthroplasty

13th March,  2011

At Malabar Palace,
Calicut, Kerala, India

Download Registration Form

For Details
Dr Anwar Marthya,
Ph:+91 9961303044

E-Mail:
anwarmh@gmail.com

 

Powered by
VirtualMedOnline

 

 

   
© Copyright of articles belongs to the respective authors unless otherwise specified.Verbatim copying, redistribution and storage of this article permitted provided no restrictions are imposed on the access and a hyperlink to the original article in Journal of Orthopaedics maintained. All opinion stated are exclusively that of the author(s).
Journal of Orthopaedics upholds the policy of Open Access to Scientific literature.