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2011, Cilt 27, Sayı 3, Sayfa(lar) 268-270 |
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DOI: 10.5146/tjpath.2011.01089 |
Auricular Angioleiomyoma: A Case Report |
Rana ÇİTİL1, Harun ÇIRALIK2, Asiye GÜL3, Hamide SAYAR2 |
1Pathology Laboratory, Kahramanmaraş State Hospital, KAHRAMANMARAŞ, TURKEY
2Department of Pathology, Sütçü İmam University, Faculty of Medicine, KAHRAMANMARAŞ, TURKEY
3Department of Otolaryngology, Kahramanmaraş State Hospital, KAHRAMANMARAŞ, TURKEY |
Anahtar Kelimeler: Anjiyoleiyomyoma, Vasküler, Leiyomyoma, Kulak |
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Anjiyoleiyomiyoma (vasküler leiyomiyoma veya anjiyomiyoma)
nadir görülen, kan damarlarının tunika mediasından köken alan
benign düz kas tümörüdür. Bu tümörler vücutta herhangi bir yerde
bulunabilir. Sıklıkla alt ekstremitede ortaya çıkmaktadır. Aurikular
anjiyoleiyomyoma oldukça nadirdir ve az sayıda olgu rapor
edilmiştir. Biz burada sol aurikular helikste anjiyoleiyomiyoması olan
38 yaşındaki bir erkek hastayı sunmaktayız. |
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Abstract
Introduction
Case Presentation
Disscussion
References
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Angioleiomyomas account of 5% of all benign neoplasms of
soft tissues. They occur mostly in the lower extremity, some
of them have been described in the head and neck region.
There are only a few cases of auricular angioleiomyoma
reported in the literature. The tumor was located over the
helix, pinna, and lobule in these reports 1-4. Hachisuga
et al. reported a large series of angioleiomyoma including
562 patients, where only 14 cases (2.8%) were in the auricle. 5,6. We describe here a 38-year-old man who presented
with an unusual lesion on the left auricular helix. |
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Abstract
Introduction
Case Presentation
Disscussion
References
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A healthy 38-year-old man presented to the ear-nose-throat
surgery clinic at our hospital with a small, intermittent
painful, slowly growing nodule located on the helix of his
left ear. He had first noted the lesion 13 years ago. No other
similar lesion was found on his face, neck and extremities.
His medical and family histories were unremarkable.
Clinical diagnosis was a vascular malformation. The
lesion was completely excised under local anesthesia.
Grossly, the specimen was an ovoid, firm, white-gray
mass, measuring 1.0x1.0x0.7 cm. The cut surface was
solid and white-gray. Microscopically, the tumor was composed of a well circumscribed proliferation of smooth
muscle bundles that surrounded numerous thick-walled
blood vessels with partially patent lumina (Figure 1).
The smooth muscle cells showed no cellular atypia or
mitoses. In immunohistochemical studies, the perivascular
proliferating smooth muscle cells and the muscular wall of thick blood vessels showed immunoreactivity for the
smooth muscle antigen (SMA). Smooth muscle stained
dark red by Masson's Trichrome (Figure 2). The endothelial
cells stained with CD34 antibody (Figure 3). Based on
these results, the tumor was diagnosed venous type
angioleiomyoma. There was no evidence of recurrence
during the six month postsurgical period.
Click Here to Zoom |
Figure 1: The tumor mass is composed of well-differentiated
smooth muscle cells and numerous thick-walled vessels with
some dilated vascular channels (H&E; x40). |
Click Here to Zoom |
Figure 2: Immunohistochemical staining for smooth muscle actin
shows a positive reaction in perivascular proliferating smooth
muscle bundles (x100). |
Click Here to Zoom |
Figure 3: The endothelial cells of the thick-walled vessels stained
with CD34 antigen (x40). |
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Abstract
Introduction
Case Presentation
Disscussion
References
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Benign tumors of smooth muscle cells can be categorized in
three clinicopathologic varieties: 1- Cutaneous leiomyomas
(pilar leiomyoma, genital leiomyoma); 2- Angiomyoma
(vascular leiomyoma or angioleiomyoma); 3- Leiomyoma
of deep soft tissue (somatic leiomyoma, gynecologic
leiomyoma) 7.
Angioleiomyomas, a subtype of leiomyomas, are rare,
benign, smooth muscle tumors that arises from tunical
media layer of small arteries and veins5. Leiomyoma was
published by Aufrecht in 1868 and it was the first report
of a case arising from blood vessels' wall. Angioleiomyoma
is classified into three histological types. 1-Capillary or
solid: the most common type, which is closely compacted
smooth muscle and many small, slit-like vascular channels.
2-Venous: thick, easily identifiable muscular walls
distinguish this type. 3- Cavernous: the vascular channels
are dilated with less smooth muscle. This is the least
common of the three types5,8.
It may occur anywhere in the skin or subcutaneous tissue.
The tumors are usually small (2 to 15 mm), solitary, round,
firm, skin-colored, and well-encapsulated1.
Hachisuga reviewed 562 cases. Among those cases, 205
cases were male and 357 were female, with a ratio of 1:1.7.
The peak incidence was between the fourth and sixth
decades5. Most common anatomical sites are lower
extremity (67%), upper extremity, head and trunk. Solid
angioleiomyoma occurs most frequently in the lower
extremity of females. The venous type is the most frequent
type found in the head and neck region usually in males,
as occurred in our case. Pain and tenderness are the
most characteristic subjective complaints in patients with
angioleiomyoma (%58)5,6. Exposure to wind, cold, local
pressure and other imperceptible stimuli cause attacks of
paroxysmal pain. Painful tumors are the most frequent type
of solid tumors. Tumors occurring in the head and neck
regions are usually not accompanied by pain5.
The clinical presentation of angioleiomyoma is nonspecific.
It is not easily differentiated from other benign subcutaneous
tumors. The diagnosis of this lesion is therefore based on
excisional biopsy3.
Grossly, the tumors are circumscribed, glistening,
white-gray nodules. Microscopically, the tumors have a
characteristic appearance that varies little from case to
case. The usual appearance is a well-demarcated nodule of
smooth muscle tissue punctuated with thick-walled vessels
with partially patent lumens. Typically, the inner layers
of smooth muscle of vessels are arranged in an orderly
circumferential fashion and the outer layers spin or swirl
away from the vessel, merging with the less well-ordered
peripheral muscle fibers. The vessels in these tumors are
difficult to classify as veins or arteries7. Various stains
have been used to identify vascular leiomyomas, including;
desmin, vimentin, actin, myosin, and Masson's trichrome.
In our patient, the tumor intensely stained with smooth
muscle actin and Masson's trichrome.
Recurrence after excision is rare5. Malignant changes
have been reported in recurrent tumors9. There is one
case report that an angioleiomyoma can occur in association
with a leiomyosarcoma10.
In this article, we describe a case of angioleiomyoma arising
from the auricle. This lesion should be considered in the
differential diagnosis of nodular masses of the auricle.
The pathological differential diagnosis must include
subcutaneus leiomyoma, hemangioma, angiofibroma,
fibromyoma, leiomyoblastoma, angiomyolipoma, vascular
leiomyosarcoma and other tumors of perivascular cells
(glomus tumors and myopericytoma)11,12. |
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Abstract
Introduction
Case Presentation
Discussion
References
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1) Inoue F, Matsumoto K: Vascular leiomyoma of the auricle. Arch
Dermatol 1983, 119:445-446 [ PubMed ]
2) Choe KS, Sclafani AP, McCormick SA: Angioleiomyoma of the
auricle: a rare tumor. Otolaryngol Head Neck Surg 2001, 125:
109-110 [ PubMed ]
3) Wang MC, Shiao AS: Auricle Angioleiomyoma. Chinese Medical
Journal (Taipei) 2002, 65:180-182 [ PubMed ]
4) Wirth GA, Sundine MJ, Kong AP, Carpenter PM: Auricular
angioleiomyoma: a case report and review of the literature. Ear
Nose Throat J 2007, 86:281-283 [ PubMed ]
5) Hachisuga T, Hashimoto H, Enjoji M: Angioleiomyoma: a
clinicopathologic reappraisal of 562 cases. Cancer 1984, 54:
126-130 [ PubMed ]
6) Ramesh P, Annapureddy SR, Khan F, Sutaria PD: Angioleiomyoma:
a clinical, pathological and radiological review. Int J Clin
Pract 2004, 58:587-591 [ PubMed ]
7) Weiss SW, Goldblum JR: Benign tumors of smooth muscle.
Enzinger & Weiss's Soft Tissue Tumors. 5th ed., Philadelphia,
Mosby Elsevier, 2008, 517-528
8) Morimoto N: Angiomyoma (vascular leiomyoma): a clinicopathologic
study. Medical Journal of Kagoshima University 1973,
24:663-683
9) Herren DB, Zimmermann A, Buchler U: Vascular leiomyoma
in an index finger undergoing malignant transformation. J Hand
Surg Br 1995, 20B:484-487 [ PubMed ]
10) Nishio J, Iwasaki H, Ohjimi Y, Ishiguro M, Kobayashi K,
Nabeshima K, Naito M, Kikuchi M: Chromosomal imbalances
in angioleiomyomas by comparative genomic hybridization. Int J
Mol Med 2004, 13:13-16 [ PubMed ]
11) Calonje E: Vascular tumors: Tumors and tumor-like conditions
of blood vessels and lymphatics. In Elder DE (Ed): Lever's
Histopathology of the Skin. 10th ed., Philadelphia, Lippincott
Williams & Wilkins, 2009, 1047-1051
12) Nall AV, Stringer SP, Baughman RA: Vascular leiomyoma of the
superior turbinate: first reported case. Head Neck 1997, 19:63-67 [ PubMed ] |
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Abstract
Introduction
Case Presentation
Discussion
References
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