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A stable swelling of the hard palateMark A. Scheper et al.Oral Surg Oral Med Oral Pathol Oral Radiol Endod2007;104:461-4

Presented by : Kush Pathak

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Contents

• Case report

• Differential Diagnosis

• Histopathological features

• Discussion

• Critical Evaluation

• Discussion

• References

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Case report• 67 year old African American male • Non painful swelling of the hard palate present for

11 year.

• Lesion was symptom free, compressible, sessile, reddish blue nodule, raised 5x5 mm and was present in the sub mucosa of the hard palate.

• Did not blanch with pressure.

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• Increase of 1mm since first reported in Jan of 1993, without change in color.

• Occlusal radiograph revealed neither change in bone nor in calcification.

• Aspiration of the lesion was negative.

• Hypertension & hypercholesteromia.

• Negative for use of smoking or alcohol.

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Differential Diagnosis

• Reactive lesions - fibroma or fibrosed pyogenic granuloma, mucocele, or lymphoid hyperplasia

• Salivary Gland Neoplasms - Pleomorphic adenoma, Monomorphic adenoma, Mucoepidermoid carcinoma, adenoid cystic carcinoma, polymorphous low-grade Adenocarcinoma, or carcinoma ex-Pleomorphic adenoma

• Mesenchymal neoplasms - fibrous histocytoma, Schwannoma, or Neurofibroma

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Reactive lesions

• Fibrosed pyogenic granuloma :

Smooth & lobulated lesion of the gingiva.

Present buccal to the dentition.

Any age.

Red, ulcerated hemorrhagic appearance-more collagenous lesion covered by intact mucosa with a normal color.

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• Traumatic fibroma : Occur due to chronic trauma.

Buccal mucosa & tongue.

Any age.

Common in women.

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• Giant Cell Variant : Younger age.

Gingiva , tongue, palate & buccal mucosa.

Greater tendency to Recur.

Symptom free, raised nodule of hard palate.

Normal color.

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• Mucocele : Any age.

Symptom free translucent blue white nodule on the lower lip.

Any oral site, minor salivary gland.

Normal in color.

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• Types :

(1)Extravasation- Characterized by disruption of the excretory duct leading to the spillage of mucin into the connective tissue.

(2) Retention- Characterized by a cystic process that develops as a result of duct obstruction.

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• Lymphoid Hyperplasia : Response to antigenic challenge.

Anywhere in oral cavity.

Palatal area involved in olders.

Female Predilection.

Discrete, firm, painless, non ulcerated, non fluctuant, slow growing mass.

Red blue to yellow orange.

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Excluded

• Vascular malformation Reddish blue lesions. Blanch - negative Needle aspiration was negative.

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Salivary Gland Neoplasms

• Benign : Most common- Pleomorphic adenoma. Monomorphic adenoma.

Less common- Basal cell adenoma. Myoepithelioma. Canalicular adenoma. Oncocytoma.

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• Pleomorphic adenoma- Minor salivary gland.

Women-3rd to 4th decade.

Symptom free

Slow growing, painless, firm single nodular mass

Smooth surface, compressible, bluish ulceration due to trauma.

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• Malignant : Mucoepidermoid carcinoma Carcinoma ex – Pleomorphic adenoma

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• Mucoepidermoid carcinoma-

Site - Palate.

Bluish raised lesion.

Resemble extravasation mucocele.

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• Mesenchymal neoplasms including Fibrous Histocytoma, Schwannoma, Neurofibroma, Leiomyoma, and Rhabdomyoma must be considered.

• Appear as asymptomatic nodular lesions, often with normal tissue coloration.

• Mesenchymal malignant lesion is excluded because of the long standing stable nature of the lesion.

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Investigations

• An excisional biopsy was performed under local anesthesia and submitted for histopathologic evaluation

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Histopathological features

• The lesion appeared macroscopically as an encapsulated soft tissue lesion that separated cleanly from the adjacent tissue.

• No evidence of bony involvement.

• Well circumscribed lesion surrounded by complete capsule.

• Containing numerous vessels.

• Surrounded by thick muscular wall..

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• Smooth muscle and collagen fibers present b/w vascular spaces.

• Muscle shows no evidence of malignant change, no capsule invaded.

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Smooth muscle origin of the tumor cells confirmed by immunohisochemical staining with smooth muscle actin.

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• Diagnosis :• Angioleiomyoma

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Discussion• Leiomyomas are uncommon benign smooth

muscle neoplasms.

• Female predilection

• Classified into 3 groups on the basis of histology : Solid leiomyoma, Angioleiomyoma, Epithelioid leiomyoma.

• Oral lesions are very rare.

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• A ratio of 2:1 benign versus malignant smooth muscle tumors of the oral cavity has been reported in the literature.

• Clinically oral angioleiomyomas occur over a wide age range of 3.5 to 85 years.

• Mean of age is 41 to 45 years.

• A male prevalence of 1.43:1 has been reported for this lesion.

• 66% affecting Caucasians and 16.1% occurring in African Americans.

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• Site : • Lip – (48.6%, with the lower lip being more

common than the upper lip)

• Palate- (21.1%, especially the hard palate)

• buccal mucosa - (9.2%)

• Tongue - (9.2%).

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• Lesions have also been reported in sulcus, floor of the mouth, gingiva.

• 9 cases of intraosseous angioleiomyomas involving mandible have also been reported.

• Soft tissue lesions appear as painless, slowly enlarging, round, elevated, sessile nodules, with variable degrees of firmness and color ranging from normal to red to blue-purple to white-gray.

• Long standing.

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• Angioleiomyomas variably firm, soft tissue nodules devoid of prominent hemorrhage.

• Histologically : Well circumscribed

With a partial to full-thickness capsule encasing numerous vascular spaces, which are surrounded by thickened muscular walls of varying size.

Between these vascular spaces is a variable mix of collagen fibers and smooth muscle fibers

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• The smooth muscle fibers exhibit characteristic eosinophilic cytoplasm and cigar-shaped nuclei.

• The confirmation of smooth muscle origin can be made immunohistochemically with antibodies to smooth muscle actin.

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• Treatment : • Complete surgical excision

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Critical Evaluation• Absence of Abstract.

• Article lacks images to the tumors explained.

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Discussion• A leiomyoma is a benign smooth

muscle neoplasm that is not premalignant. They can occur in any organ, but the most common forms occur in the uterus, small bowel and the esophagus.

• A leiomyoma of the uterus is commonly called a fibroid.

• Classified into 3 groups on the basis of histology : Solid leiomyoma, Angioleiomyoma, Epithelioid leiomyoma.

• Oral Leiomyomas are uncommon smooth muscle tumors due to the scant presence of this tissue in the oral cavity.

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Multiple cutaneous leiomyomas in cluster on the upper back.

Multiple interlacing bundles of smooth muscle fibers in the dermis

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Bundle of smooth muscle

Normal collagen

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• Angioleiomyoma : A benign tumor composed of mature smooth muscle and vascular channels, occurring in the sub cutis or deep dermis.

• Most common type.

• Angioleiomyomas usually are asymptomatic tumors that can become malignant.

• They are common, typically painful, benign neoplasm that originate from smooth muscle cells of arterial or venous walls and contains thick-walled vessels.

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• Angioleiomyoma is often associated with pain. Pain is probably mediated by nerves present in the tumor and in the capsule due to either mechanical stretching or through mast cell mediation.

• Other painful tumors of soft tissue and skin are glomus tumor, eccrine spiradenoma, angiolipoma and traumatic neuroma.

• Site:  These are usually located in the sub cutis and

deep dermis of the extremities, particularly the lower leg.

Cases have been reported on the head and upper trunk and these are often painless.

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Angioleiomyoma in Gingiva

Click icon to add picture

Bulging out mass 2×3 cm in size over right lower posterior lingual gingival withnormal mucosal appearance.

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No obvious bony destruction on panoramic film over right side posterior mandibleAngioleiomyoma in right lingual gingiva – A case report. Kai-Feng Hung;

Chin J Oral Maxillofac Surg 16: 179-187, September 2005

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17×21 mm, ill-circumscribed, nonhomogeneous solid mass in the right submandibular triangle attached to lingual surface of mandible.

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Proliferation of spindle-shape cellsinterspersed among multiple vascular spaces of varied size in sub mucosal connective tissueof the gingiva.

Thickness of blood vessel walls composed with smoothmuscle cells in a whir ling pattern. eosinophilic cytoplasmand spindled shape vesicular nuclei

Angioleiomyoma in right lingual gingiva – A case report. Kai-Feng Hung; Chin J Oral Maxillofac Surg 16: 179-187, September 2005

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• Angioleiomyoma could be separated into three histologic subtypes:

Solid or capillary - Accounts for most cases on the legs. Vascular channels are numerous but small and slit-like.

Cavernous - Accounts for most cases on the arms. The dilated vascular channels lack distinct muscular walls.

Venous - Accounts for most cases on the head. The vascular channels have thick muscular walls.

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Immunohistochemistry• Indicates that the smooth muscle cells express

vimentin, desmin and smooth muscle actin.

Smooth muscle Actin (SMA)- Positive Smooth muscle neoplasms Myoepithelial cells in breasts, salivary glands and

sweat glands.    

A monoclonal antibody against alpha-smooth muscle actin: a new probe for smooth muscle differentiation. Skalli O et al. J

Cell Biol. 1986 Dec;103(6 Pt 2):2787-96.

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Desmin - Positive Identification of smooth muscle and skeletal

muscle tumors. Differentiation of reactive mesothelial cells

(positive) from mesothelioma (negative) and carcinoma (negative) in effusions and histological specimens

Vimentin – Positive Mesenchymal cells

Collage type IV – Positive

HMB-45- Negative Monoclonal antibody that identifies Immature

melanosomes

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• Prognosis – Excision is almost curative with rare recurrence.

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• Five hundred sixty-two cases of angioleiomyoma on soft tissues were reappraised clinicopathologically.

• There was a preponderance in females with a ratio of 1.7:1.

• The angioleiomyoma could be separated into three histologic subtypes: capillary or solid (374 cases), cavernous (61 cases), and venous (127 cases) types.

• Five hundred (89%) occurrences were in the extremities, 48 in the head, and only 14 in the trunk

Angioleiomyoma. A clinicopathologic reappraisal of 562 cases. Hachisuga T, Hashimoto H, Enjoji M. Cancer. 1984

Jul 1;54(1):126-30.

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• The tumors caused pain and/or tenderness in 327 cases, in which 262 were of the solid type.

• In 16 cases, small groups of mature fat cells were demonstrated within the tumor, suggesting the hamartomatous nature of these smooth muscle tumors.

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• A 67-year-old man without a personal history of interest was referred to the department for evaluation of a lingual tumor for the last 6 months that was asymptomatic but grew progressively.

• Mass was located in the left anterior third of the tongue. The largest diameter of the tumor was approximately 5 mm and the tumor was of hard consistency and not adhered to deep planes

Angioleiomyoma lingual –A case report. I. Peña González et al. Rev Esp Cir Oral y Maxilofacial 2008;30,2 (marzo-

abril):101-105

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• Lingual mucosa located over the tumor had a normal appearance.

• No lymph node enlargement

• Excisional biopsy was performed.

• H-E staining revealed tissue characterized by proliferation of small-caliber blood vessels with round Lumina and thick walls.

• Acidophilic spindle cells present.

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• IHC study was made to establish the type of cells that constituted the tumoral stroma.

• Common muscle actin yielded a positive result.

• Then, to identify the muscle strain, the tissue was stained with smooth muscle actin, which gave an intensely positive result.

• This established the diagnosis of angioleiomyoma and ruled out malignancy in light of the absence of cellular atypia and the scant number of mitoses.

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Blood vessel with thick walls surrounded by acidophilicspindle cells.

Staining with smooth-muscle actin-specific antibodies

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• 38-year-old woman was referred for treatment of a suspected sarcoma over her right lower posterior lingual gingiva.

• It was 2x3 cm in size with no pus discharge.

• Spindle shaped cells were seen.

• Thickened blood vessels

• Eosinophilic cytoplasm

Angioleiomyoma in Right Lingual Gingiva – A Case Report; Kai-Feng Hung et al. Chin J Oral Maxillofacial Surg 16: 179-187, September 2005

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Special immunohistochemical stains of HHF-35 demonstrated focal positive results over these spindle cells

MIB-1 special staining showed about 15% of mitotic activity rate

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• 57 year old presented with a chief complaint of a growth in his lower left back tooth region of 15 days duration.

• Measuring 3 cm anterioposteriorly, 1.5 cm mediolaterally.

• Asymptomatic and without any discharge.

• Excisonal biopsy was performed of the lesion along with extraction of teeth 37, 36.

• H & E sections showed numerous dilated blood vessels.

Recurrent oral angioleiomyoma. VG Mahima, Karthikeya Patil, HS Srikanth ;

Contemporary clinical dentistry. April-june 2011; volume 2 : issue 2

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• Smooth muscle cells had well defined ‘ CIGAR SHAPED’ nuclei.

• Various amount of collagen was found to be interspersed between vascular spaces.

• Special staining was performed using Mason’s trichrome, which stained smooth muscle cells red and collagen fibers blue.

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• After two months of the excision the patient reported with similar growth in the same location that had started 10 days prior to his reporting and had rapidly increased in size.

• Clinical presentation was same

• Excisional biopsy and histopathological examination confirmed that it was angioleiomyoma

• There has been no further recurrence, 32 months following treatment.

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• 3 cases were reported with nodular overgrowth over lip from last 6 months, 19 years and 5 months respectively.

• Histopathological examination revealed a well circumscribed smooth muscle proliferation, with spindle shaped cells and blood vessels and large vascular spaces.

• Eosinophilic cytoplasm seen.

• Smooth muscle showing interlacing pattern with collagen fibers between dilated and slit-like vascular spaces.

Angioleiomyoma affecting the lips: Report of 3 cases and review of the literature ; Luiz-Alcino Gueiros et al: Med Oral Patol Oral Cir Bucal. 2011 Jul 1;16 (4):e482-7.

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• Immunohistochemically, in al the three cases alpha smooth muscle actin, desmin and HHF-35 were positive.

• CD34 was also positive on the endothelial cells.

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References Angioleiomyoma in right lingual gingiva – A case

report. Kai-Feng Hung; Chin J Oral Maxillofac Surg 16: 179-187, September 2005

Angioleiomyoma. A clinicopathologic reappraisal of 562 cases. Hachisuga T, Hashimoto H, Enjoji M. Cancer. 1984 Jul 1;54(1):126-30.

Evaluation of Biological Potential of smooth muscle tumors. Miettinen M, Fetsch JF. Histopathology. 2006 Jan;48(1):97-105

A monoclonal antibody against alpha-smooth muscle actin: a new probe for smooth muscle differentiation. Skalli O et al. J Cell Biol. 1986 Dec;103(6 Pt 2):2787-96.

Recurrent oral angioleiomyoma. VG Mahima, Karthikeya Patil, HS Srikanth ; Contemporary clinical dentistry. April-june 2011; volume 2 : issue 2

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Angioleiomyoma. A clinicopathologic reappraisal of 562 cases. Hachisuga T, Hashimoto H, Enjoji M. Cancer. 1984 Jul 1;54(1):126-30.

Angioleiomyoma lingual –A case report. I. Peña González et al. Rev Esp Cir Oral y Maxilofacial 2008;30,2 (marzo-abril):101-105

Angioleiomyoma affecting the lips: Report of 3 cases and review of the literature ; Luiz-Alcino Gueiros et al: Med Oral Patol Oral Cir Bucal. 2011 Jul 1;16 (4):e482-7.

A very rare form of leiomyoma: Mandible angioleiomyoma. Hüseyin Koca et al. International journal of pediatric otorhinolaryngology. June 2006; 110 -14

http://e-immunohistochemistry.info/web/Angioleiomyoma.htm

http://e-immunohistochemistry.info/web/Antigens/desmin.htm

Wikipedia.org

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