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Leiomyoma of the oral cavity
ORAL
PATHOLOGY
General
Section
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.
.
.
.
.
.
.
.
.
.
.
.
.
.
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.
.
...*..
LEIOMYOMA OF THE ORAL CAVITY Review of the Literature and Report of a Case D. N. Hngy, D.D.S.,” Victor Halperh, D.D.S.,“* Carroll Wood III, D.D.X.,““” New Orleans, La. 1~1yola
Trnicersity,
School
and
of Ikmtistry
may occur anywhere in the human body that smooth muscle is present. The uterus is the most common site, but these tumors arc also of relatively frequent occurrence in the wall of the alimentary tract, in t,he skin, and in subcutaneous tissue. They are rare in the oral cavity, as indicated by the few reported cases. A feasible explanation for this is the paucity of smooth muscle in the oral cavity as compared with the abundant sources in the ot,her areas. In the skin, for example, there is smooth muscle tissue in the arrectores pilorum muscles, the walls of blood vessels, and the muscular apparatus in the depths of the skin. Thr latter as a source of smooth muscle for the l&myoma is support,ed by the significant number of these tumors that havcxoccurred in regions where this special muscle is present. This special muscle apparatus includes t,hr muscularis sexualis, smooth muscle in the corium or supcrficial subcutaneous tissue of the scrotum, penis, and labium majus; the muscnIaris mammillac et arcola in the nipple and areola; and musculi cutis diagonalcs in the corium OF the extensor aspects of the extremities and in the cheek, scalp, and forrhead.” Probably the chief source of smooth muscle in the oral cavity is the tunica media of the walls of blood vessels. According to Schumacher, as reported b> Stout, Schaffer observed that smooth muscle was frequently present in the cirrumvallate papillae of the tongue, and this has been alleged by some authors t,o be a source.*. FOther sources that have been mentioned are the ductus lingualis and displaced cmbr.vonal tissue.4.6 EIOMYOMAS
L
Present
*Formerly address: **Professor ***Formerly
Assistant Professor of Oral Pathology. 2720 Capitol Ave., Sacramento, Calif. of Oral Pathology, Loyola University, Senior Resident, Department of Oral
748
Loyola
University,
School of Surgery.
Dentistry. Charity
School Hospital.
of
Dentistry.
Volume Number
LEIOMYOMA
17 6
HISTORY
AND
REVIEW
OF
THE
OF
ORAL
CAVITY
749
LITERATURE
As early as 1854 Virchow reported a case in which a group of small tumors, composed of smooth muscle, involved the skin of the thoracic wall near the areolar tissue. Since that time numerous cases of multiple and solitary leiomyomas, occurring in such areas as the scrotum, labium majus, nipple, and extensor surfaces of the extremities have been reported. An interesting and relatively frequent finding in these lesions was pain of the paroxysmal type; this was attributed to contraction of the smooth muscle.” Before Stout reported the first leiomyoma of the oral cavity in the English literature in 1938, five cases had been reported in the foreign literature. The first case was reported by Blanc1 in 1884. In that case the tumor occurred in a 33-year-old man and was located at the base of the tongue in the tonsillar region. The tumor was rather large and projected from the surface. It was asymptomatic except for a change in the patient’s voice which had been noticed by other people for approximately 12 years. Part of the lesion was removed with a wire. This was followed by infection, necrosis, and a sloughing of the mass. There was no evidence of recurrence after 5 months. Microscopic examination showed the tumor to be comprised of smooth muscle and abundant fibrous tissue. In 1905 Glas4 reported a case involving the tongue of a 44-year-old man. This t,umor located near the foramen cecum, was described as being the size of a walnut and without symptoms other than a scratchy sensation in the throat. The tumor was biopsied, and microscopic examination showed it to be composed of bundles of smooth muscle fibers, fibrous tissue, and some mucus glands. The tumor was removed with the galvanic cautery, and healing was normal; however, the patient was not followed for possible recurrence. Because of its location, Glas suggested that this tumor might have had its origin from the ductus lingualis. He also mentioned Schaffer’s sometimes finding smooth muscle in the circumvallate papillae and suggested this as a possible origin for the tumor reported by Blanc. Fein,3 in 1905, reported a case in a 20-year-old woman. It was discovered when the patient sought treatment for a pharyngitis which had caused hoarseness for several days. The tumor was described as a small, pale, pedunculated lesion projecting off the left posterior border of the soft palate. The patient stated that she had noticed the lesion in a mirror some time previously but, because of the lack of symptoms, had decided that it was a normal occurrence. The tumor was removed with a cold loop and submitted for microscopic examination. Dr. Sternberg examined the tissue and described it as being comprised of interlacing bundles of smooth muscle fibers, but there was no mention of blood vessels. Fein expressed the opinion that this tumor could have had its origin either from a blood vessel or from displaced embryonal tissue. Weil,l” in 1914, reported a leiomyoma that occurred on the posterior surface of the uvula in a 69-year-old man. He described the tumor as being the size and shape of half a plum and pedunculated. The patient had been aware of the lesion, which was asymptomatic, for a number of years. The tumor was
1884
1905
1905
1914
1937
1937
1937
1944
1963
--.
3. Glas
3. Fein
4. Weil
5. stout
6. Stout
Peter
9. Hagy
.---..
i.
8. Hurford, Ackerman, and Robinson
et al.
1. Blanc
AUTHORS
.-
surface
Posterior
69
50
Male
Male
E’cmalc
..-.. -------..
76
------ -----.- ----
Lip
3' st1c
Floor _ of mouth,
40
Female
left
pole of sub salivary gland
Posterior lingual
29
Female
Mall?
left
of
Lateral surface of tongue, halfway bet,ween tip and epiglottis
Base of tongue, side
uvula
lrorder
Posterior palate
20
Female
of soft
Median terminal sulcus of tongue, near foramen recum
44
Male
of tongue
LOCATION
Base
AGE (YEARS)
1. LEIOMYOMAS
33
Male
PATIENT’S SEX
TABLE
/
followup
follo\r-up
follow-up
after
after
6
5
CAYITY
30 months a mass _. noted 111 tlirs area, and recurrence was suggested but not confirmed was
After
Ku’0 follow-up
No recurrence years
Ko
so
No
ORAL
FOLl.OW-K:P
THE
No recurrence months
OF
I
Xonvascular
MICROSCOPIC TYPE
---.
__I
Circumrallate
POSSIBLE ORIGIN
papillae
-1 2,
Volume Number
17 6
LEIOMYOMA
OF
ORAL
CAVITY
751
removed by electrocautery and the tissue was submitted for microscopic study to Koritschoner, who diagnosed it as a leiomyoma with large vascular spaces. In I937 Peter reported a case that was associated with the sublingual gland and extended into the floor of the mouth. This tumor occurred in a JOyear-old woman.8 Stout’ reported two cases in 1938 and gave a very comprehensive review of the literature. One of his cases involved a 50-year-old man, and the tumor was located on the lateral surface of the base of the tongue and projected between it and the epiglottis. On examination, the lesion was found to be soft, fluctuant, and movable and, clinically it was thought to be an angioma or a cyst. The only symptom was a sore throat of 5 weeks’ duration. The lesion was removed with a snare, and Stout described it as being composed of fibrous tissue and smooth muscle. Thick-walled blood vessels were present throughout the tumor. There was no recurrence after 6 years. Stout’s’ second case was that of a 29-year-old woman with a tumor on the lateral border of the tongue between the tip and the epiglottis. It was soft, pedunculated, and asymptomatic. The tumor was excised, and the wound healed normally. Microscopically, the tumor was composed of a large amount of fibrous tissue and normal-appearing smooth muscle that had no relationship with blood vessels. The eighth case was that reported by Burford, Ackerman, and Robinson2 in 1944. The tumor occurred in a 32-year-old Negro man and was located in the floor of the mouth on the right side. The patient had complained of difficulty in swallowing, and a clinical examination revealed a rather large, grayish white, firm, irregular mass growing out of the right side of the floor of the mouth. A biopsy was performed, and microscopic examination showed t,hat the tumor was made up mostly of connective tissue although smooth muscle and numerous blood vessels were also present. The diagnosis was leiomyoma, and the mass was surgically excised under endotracheal ether anesthesia. A recurrence was suspected when a follow-up examination was made 20 months later, and further surgical intervention was anticipated. Summarizing the pertinent findings in these cases (Table I), we find that patients ranged in age between 20 and 69 years; five were men and three were women. The tongue was the most common site, with three tumors occurring at the base and one on the lateral border halfway between the tip and the epiglottis. As for other sites, one tumor was found on the posterior surface of the soft palate, one occured on the posterior surface of the uvula, and two were located in the floor of the mouth (one associated with the sublingual salivary gland and one with the t,ongue). With regard to clinical characteristics, all the tumors were superficial and three were pedunculated. They were firm, movable, and bluish or reddish in color, depending on their depth and vascularity. They were asymptomatic except for the patient’s awareness that a mass was present. Stout has expressed his opinion concerning the origin of these tumors. He states that smooth muscle is sometimes found in the circumvallate papillae and that in Blanc’s and Glas’s cases the tumors probably had their origin in this tissue. He suggested that in his second case and in Fein’s case the
Fig. I. Fig. Fig. l.-The majority of the cells are rounded or polygonal. rather elongated, blunt-ended shape of differentiated leiomyoblast. (Hematoxylin niflcation, Xl00 : reduced s.) Fig. %.-Rounde~l or polygonal cells, some of which have R clear nucleus. (Hematoxylin and eosin stain. Magnification, X130 ; rr~luccvl $,.)
2. than Imving the usw I and eosin stain. Xrrzonfx
surrounding
tllv
tumors were dq’sontogcnr~tic in origin. Thcl vascular tumors in the c’ast’s IVported by Weil and h\- Burford and associates and in Stout,‘s first case were all assumed to hnvc had their origin in ;I blood vessel wall.” C.iSE
REPORT
.Y 76.year-old Negro woman was seen in the lkntal Clinic at Charity Hospital on April 1, 1963, for treatment of a %ump on her lip. ” The lesion appeared as a painless, firm, round, raiseiI nodule, approximately 2 cm. in diameter. The overlying mucosa was slightly darker, and thr clinical impression was mucowle. Routine laboratory work was not done. The patient st,ated that she had ljeen in good healt,h with the exception of arthritis for approximately 20 years. TTnder local Monocainc anesthesia, an invision was made and the lesion, which separatwl was removed. The specimen was submitted to the wry easily from the surrounding tissue, Pathology Department for histopathologic examination. Pntkologic Findings.-The gross specimen was a spherical nodule of tan tissue, 1.0 WL. in diameter. The cut surface revealed a homogenous white tissue with two foci of gray gelatinous material. Microscopically, the tumor was comprised chiefly of bizarre round cells, some of which had clear zones surrounding the nucleus. In some areas there were elongated cells which rcscmbled ordinary smooth muscle cells. Masson’s trichrome stain failed to demonstrate myofihrils, but coarse granules w+rP noted in the cytoplasm. Xicroscopkc diagnosis: Atypical leiomyoma.* Tulane
*The microscopic diagnosis was University School of Medicine,
made New
by Dr. Orleans,
W. H. La.
Sternberg,
Professor
of Pathology,
Volume Number
17 6
LEIOMYOMA
OF
ORAL
CAVITY
Fig. 3. Fig. 3.-An area in which some of the tumor cells are appearance of a leiomyoblast. (Hematoxylin and eosin stain. Fig. 4.-An area showing a blood vessel which is thought toxylin and eosin stain. Magnification, X430 ; reduced $5.)
753
Fig. 4. elongated, suggesting more usual Magnification, X430 ; reduced ‘$6.) to be purely nutritional. (Hema-
DISCUSSIOh‘
Microscopically, these tumors have been divided into a vascular and a nonvascular type. The vascular type has been described as containing numerous thick-walled vessels with small rounded or stellate lumina. Although some cases of subcutaneous leiomyomas have definitely been shown to arise from the walls of veins, there has been some controversy in most cases as to whether the vessels are arteries or veins. Stout” observed that the vessels in these cases were atypical and not identifiable as either arteries or veins but bore more resemblance to veins. We would like to point out that the vascular types in t,he oral cavity have been thought to be derived from the smooth muscle of blood vessels. The nonvascular type is not without blood vessels but contains only a few, which are purely nutritional. Recently Stout reported a group of unusual smooth muscle tumors that are of relatively common occurrence in the stomach. He stated that, because of their bizarre microscopic appearance, these tumors have puzzled the majority of the pathologists and that it is not suprising that they have been incorrectly neurofibromas, neurilemmomas, glomus diagnosed as fibromas, fibrosarcomas, and myriad other entities. He described tumors, granular-cell myoblastomas, the lesions as being comprised predominately of rounded or polygonal cells,
only a. few of whicah c~ontaincd (~longatctl l)lunt-cqrded nuclei that co~rltl 1)~ idcntificd as nl~cl~i ot’ I(~iorrr~o})lwsts. Thch rounded cells cxhibitc,d a nliltll~~ acidophilic cytoplasm, tllv al~scmc ol’ Irlyofihrils, and oftw a ~lcar zone partI!or completely surrounding the nuclt7w. (‘linically and grossly~ these lesions were not unlike the leiomyomas that OWUI’ in other locations. dlthough thr qc>at majority wcw c~omplt~tIely bc&gn in their behavior, two out of the sixty-nincb cases reported proved their malignancy by mrtastasizing. Stout attempted to establish criteria for determining their behavior. The most significant findin g was thcl high mitotic rate in isolated zones of thr two tumors that met~wstasizcd. There wvc~rc nineteen rnitoses for one case ant1 thirteen for the other in fifty high-power fields, which was very high when compared to the remaining sixty-seven tumors. Stout stated that, the only other locations in which he had observed these unusual smooth rnusclc tumors were the intestinal tract and the uterus. Because of the bizarre appcarancc and the rnalignant behavior of a few of these tumors, Stout) oxpressed his opposition to calling them leiomyomas and suggested the term hizcrrw leiomyoblostomcl.’ SUMMARY
AKD
COSCLUSION
We have prtrscnt,ed a case of leiomyoma of the lip and a review of the lit’crature. Onlv eight cases involving the oral cavity hare been reported up to this date, and all have been rc1ativel.v uniform in their clinical and microscopic characteristics. They were all observed to contain fairly well-differentiated smoot,h muscle cells. The case prcscnted here is similar in its clinical features to the previously reported cases but, differs in its microscopic apprarante. Stout agrees that this tumor resembles a group of unusual smooth rriusclc tumors which he has rccc>ntly observed in the stomach. Because most of thcsc tumors were benign, although a small percentage showed malignant behavior, Stout considered it wrong to call them either leiomyomas or leiornyosarcomas. He suggested the terrn leiomyoblastoma, as it denotes neither benignancy nor malignancy. As mentioned previously, the tumors t,hat, showed rnalignant bchavior differed from the benign ones only in the number of mitoses. In this present ease Stout was able to find only two rnitoses in fiftv high-power fields, which he said was a good indication that the tumor was benign.s Of COU~SC, WC cannot be sure of the origin of this smooth muscle tumor, but two possible SOUTCW are (1) the wall of a blood vessel and (2) displaced rmbryonal tissue. The fact that this lesion is not of the vascular type, does not preclude its origin from a blood ~sscl wall. REFERESCES
1. Blanc, 2. Burford,
E.:
Travaux originaux, Gaz. hcbd. de mPd. et chir. 21: Fll-613, 1884. IV. N., Ackerman, T,. V., and Robinson, H. R. G.: T,eiomyoma of t.he Tongue, Case 20, R. M. Efsch No. 4293, Am. .J. Orthodontics & Oral Surg. 30: 395397, 1944. 3. E’ein, .J.: Ein Leiomyom des Gaumens, Arch. Laryng. u. Rhin. 17: 533-535, 1905. 4. Glas, E.: Beitrage zur Patholoyie der Zllnffenffroundturnoren, Wien. klin. Wchnschr. 18:
5. stout,
746-752.
1905.
A. P.: golitarp 435-469, 1937.
Cutaneous
and
Huhcutaneous
Leiomyoma,
Am.
J.
Cancer
29:
Volume 17 Number6 G. 7. 8. 9.
LEIOMYOMA
OF
ORAL
CAVITY
755
Stout, A. P.: Leiomyoma of the Oral Cavity, Am. J. Cancer 34: 31-36, 1938. Stout, A. P.: Bizarre Smooth Muscle Tumors of the Stomach, Cancer 15: 400-409, 1962. Stout, A. P.: Personal Communication, May, 19G3. Thoma, K. H., and Goldman, H. M.: Oral Pathology, ed. 5, St. Louis, 1960, The C. V. Mosby Company, p. 1188. 10. Weil, I.: Ein Leiomyom der Uvula, Monatschr. Ohrenh. 48: 1002, 1914; Zentralbl. f. Laryngol. 31: 203, 1915.
PATHOLOGY
General
Section
.
.
.
.
.
.
.
.
.
.
.
.
.
.
.
.
.
.
...*..
LEIOMYOMA OF THE ORAL CAVITY Review of the Literature and Report of a Case D. N. Hngy, D.D.S.,” Victor Halperh, D.D.S.,“* Carroll Wood III, D.D.X.,““” New Orleans, La. 1~1yola
Trnicersity,
School
and
of Ikmtistry
may occur anywhere in the human body that smooth muscle is present. The uterus is the most common site, but these tumors arc also of relatively frequent occurrence in the wall of the alimentary tract, in t,he skin, and in subcutaneous tissue. They are rare in the oral cavity, as indicated by the few reported cases. A feasible explanation for this is the paucity of smooth muscle in the oral cavity as compared with the abundant sources in the ot,her areas. In the skin, for example, there is smooth muscle tissue in the arrectores pilorum muscles, the walls of blood vessels, and the muscular apparatus in the depths of the skin. Thr latter as a source of smooth muscle for the l&myoma is support,ed by the significant number of these tumors that havcxoccurred in regions where this special muscle is present. This special muscle apparatus includes t,hr muscularis sexualis, smooth muscle in the corium or supcrficial subcutaneous tissue of the scrotum, penis, and labium majus; the muscnIaris mammillac et arcola in the nipple and areola; and musculi cutis diagonalcs in the corium OF the extensor aspects of the extremities and in the cheek, scalp, and forrhead.” Probably the chief source of smooth muscle in the oral cavity is the tunica media of the walls of blood vessels. According to Schumacher, as reported b> Stout, Schaffer observed that smooth muscle was frequently present in the cirrumvallate papillae of the tongue, and this has been alleged by some authors t,o be a source.*. FOther sources that have been mentioned are the ductus lingualis and displaced cmbr.vonal tissue.4.6 EIOMYOMAS
L
Present
*Formerly address: **Professor ***Formerly
Assistant Professor of Oral Pathology. 2720 Capitol Ave., Sacramento, Calif. of Oral Pathology, Loyola University, Senior Resident, Department of Oral
748
Loyola
University,
School of Surgery.
Dentistry. Charity
School Hospital.
of
Dentistry.
Volume Number
LEIOMYOMA
17 6
HISTORY
AND
REVIEW
OF
THE
OF
ORAL
CAVITY
749
LITERATURE
As early as 1854 Virchow reported a case in which a group of small tumors, composed of smooth muscle, involved the skin of the thoracic wall near the areolar tissue. Since that time numerous cases of multiple and solitary leiomyomas, occurring in such areas as the scrotum, labium majus, nipple, and extensor surfaces of the extremities have been reported. An interesting and relatively frequent finding in these lesions was pain of the paroxysmal type; this was attributed to contraction of the smooth muscle.” Before Stout reported the first leiomyoma of the oral cavity in the English literature in 1938, five cases had been reported in the foreign literature. The first case was reported by Blanc1 in 1884. In that case the tumor occurred in a 33-year-old man and was located at the base of the tongue in the tonsillar region. The tumor was rather large and projected from the surface. It was asymptomatic except for a change in the patient’s voice which had been noticed by other people for approximately 12 years. Part of the lesion was removed with a wire. This was followed by infection, necrosis, and a sloughing of the mass. There was no evidence of recurrence after 5 months. Microscopic examination showed the tumor to be comprised of smooth muscle and abundant fibrous tissue. In 1905 Glas4 reported a case involving the tongue of a 44-year-old man. This t,umor located near the foramen cecum, was described as being the size of a walnut and without symptoms other than a scratchy sensation in the throat. The tumor was biopsied, and microscopic examination showed it to be composed of bundles of smooth muscle fibers, fibrous tissue, and some mucus glands. The tumor was removed with the galvanic cautery, and healing was normal; however, the patient was not followed for possible recurrence. Because of its location, Glas suggested that this tumor might have had its origin from the ductus lingualis. He also mentioned Schaffer’s sometimes finding smooth muscle in the circumvallate papillae and suggested this as a possible origin for the tumor reported by Blanc. Fein,3 in 1905, reported a case in a 20-year-old woman. It was discovered when the patient sought treatment for a pharyngitis which had caused hoarseness for several days. The tumor was described as a small, pale, pedunculated lesion projecting off the left posterior border of the soft palate. The patient stated that she had noticed the lesion in a mirror some time previously but, because of the lack of symptoms, had decided that it was a normal occurrence. The tumor was removed with a cold loop and submitted for microscopic examination. Dr. Sternberg examined the tissue and described it as being comprised of interlacing bundles of smooth muscle fibers, but there was no mention of blood vessels. Fein expressed the opinion that this tumor could have had its origin either from a blood vessel or from displaced embryonal tissue. Weil,l” in 1914, reported a leiomyoma that occurred on the posterior surface of the uvula in a 69-year-old man. He described the tumor as being the size and shape of half a plum and pedunculated. The patient had been aware of the lesion, which was asymptomatic, for a number of years. The tumor was
1884
1905
1905
1914
1937
1937
1937
1944
1963
--.
3. Glas
3. Fein
4. Weil
5. stout
6. Stout
Peter
9. Hagy
.---..
i.
8. Hurford, Ackerman, and Robinson
et al.
1. Blanc
AUTHORS
.-
surface
Posterior
69
50
Male
Male
E’cmalc
..-.. -------..
76
------ -----.- ----
Lip
3' st1c
Floor _ of mouth,
40
Female
left
pole of sub salivary gland
Posterior lingual
29
Female
Mall?
left
of
Lateral surface of tongue, halfway bet,ween tip and epiglottis
Base of tongue, side
uvula
lrorder
Posterior palate
20
Female
of soft
Median terminal sulcus of tongue, near foramen recum
44
Male
of tongue
LOCATION
Base
AGE (YEARS)
1. LEIOMYOMAS
33
Male
PATIENT’S SEX
TABLE
/
followup
follo\r-up
follow-up
after
after
6
5
CAYITY
30 months a mass _. noted 111 tlirs area, and recurrence was suggested but not confirmed was
After
Ku’0 follow-up
No recurrence years
Ko
so
No
ORAL
FOLl.OW-K:P
THE
No recurrence months
OF
I
Xonvascular
MICROSCOPIC TYPE
---.
__I
Circumrallate
POSSIBLE ORIGIN
papillae
-1 2,
Volume Number
17 6
LEIOMYOMA
OF
ORAL
CAVITY
751
removed by electrocautery and the tissue was submitted for microscopic study to Koritschoner, who diagnosed it as a leiomyoma with large vascular spaces. In I937 Peter reported a case that was associated with the sublingual gland and extended into the floor of the mouth. This tumor occurred in a JOyear-old woman.8 Stout’ reported two cases in 1938 and gave a very comprehensive review of the literature. One of his cases involved a 50-year-old man, and the tumor was located on the lateral surface of the base of the tongue and projected between it and the epiglottis. On examination, the lesion was found to be soft, fluctuant, and movable and, clinically it was thought to be an angioma or a cyst. The only symptom was a sore throat of 5 weeks’ duration. The lesion was removed with a snare, and Stout described it as being composed of fibrous tissue and smooth muscle. Thick-walled blood vessels were present throughout the tumor. There was no recurrence after 6 years. Stout’s’ second case was that of a 29-year-old woman with a tumor on the lateral border of the tongue between the tip and the epiglottis. It was soft, pedunculated, and asymptomatic. The tumor was excised, and the wound healed normally. Microscopically, the tumor was composed of a large amount of fibrous tissue and normal-appearing smooth muscle that had no relationship with blood vessels. The eighth case was that reported by Burford, Ackerman, and Robinson2 in 1944. The tumor occurred in a 32-year-old Negro man and was located in the floor of the mouth on the right side. The patient had complained of difficulty in swallowing, and a clinical examination revealed a rather large, grayish white, firm, irregular mass growing out of the right side of the floor of the mouth. A biopsy was performed, and microscopic examination showed t,hat the tumor was made up mostly of connective tissue although smooth muscle and numerous blood vessels were also present. The diagnosis was leiomyoma, and the mass was surgically excised under endotracheal ether anesthesia. A recurrence was suspected when a follow-up examination was made 20 months later, and further surgical intervention was anticipated. Summarizing the pertinent findings in these cases (Table I), we find that patients ranged in age between 20 and 69 years; five were men and three were women. The tongue was the most common site, with three tumors occurring at the base and one on the lateral border halfway between the tip and the epiglottis. As for other sites, one tumor was found on the posterior surface of the soft palate, one occured on the posterior surface of the uvula, and two were located in the floor of the mouth (one associated with the sublingual salivary gland and one with the t,ongue). With regard to clinical characteristics, all the tumors were superficial and three were pedunculated. They were firm, movable, and bluish or reddish in color, depending on their depth and vascularity. They were asymptomatic except for the patient’s awareness that a mass was present. Stout has expressed his opinion concerning the origin of these tumors. He states that smooth muscle is sometimes found in the circumvallate papillae and that in Blanc’s and Glas’s cases the tumors probably had their origin in this tissue. He suggested that in his second case and in Fein’s case the
Fig. I. Fig. Fig. l.-The majority of the cells are rounded or polygonal. rather elongated, blunt-ended shape of differentiated leiomyoblast. (Hematoxylin niflcation, Xl00 : reduced s.) Fig. %.-Rounde~l or polygonal cells, some of which have R clear nucleus. (Hematoxylin and eosin stain. Magnification, X130 ; rr~luccvl $,.)
2. than Imving the usw I and eosin stain. Xrrzonfx
surrounding
tllv
tumors were dq’sontogcnr~tic in origin. Thcl vascular tumors in the c’ast’s IVported by Weil and h\- Burford and associates and in Stout,‘s first case were all assumed to hnvc had their origin in ;I blood vessel wall.” C.iSE
REPORT
.Y 76.year-old Negro woman was seen in the lkntal Clinic at Charity Hospital on April 1, 1963, for treatment of a %ump on her lip. ” The lesion appeared as a painless, firm, round, raiseiI nodule, approximately 2 cm. in diameter. The overlying mucosa was slightly darker, and thr clinical impression was mucowle. Routine laboratory work was not done. The patient st,ated that she had ljeen in good healt,h with the exception of arthritis for approximately 20 years. TTnder local Monocainc anesthesia, an invision was made and the lesion, which separatwl was removed. The specimen was submitted to the wry easily from the surrounding tissue, Pathology Department for histopathologic examination. Pntkologic Findings.-The gross specimen was a spherical nodule of tan tissue, 1.0 WL. in diameter. The cut surface revealed a homogenous white tissue with two foci of gray gelatinous material. Microscopically, the tumor was comprised chiefly of bizarre round cells, some of which had clear zones surrounding the nucleus. In some areas there were elongated cells which rcscmbled ordinary smooth muscle cells. Masson’s trichrome stain failed to demonstrate myofihrils, but coarse granules w+rP noted in the cytoplasm. Xicroscopkc diagnosis: Atypical leiomyoma.* Tulane
*The microscopic diagnosis was University School of Medicine,
made New
by Dr. Orleans,
W. H. La.
Sternberg,
Professor
of Pathology,
Volume Number
17 6
LEIOMYOMA
OF
ORAL
CAVITY
Fig. 3. Fig. 3.-An area in which some of the tumor cells are appearance of a leiomyoblast. (Hematoxylin and eosin stain. Fig. 4.-An area showing a blood vessel which is thought toxylin and eosin stain. Magnification, X430 ; reduced $5.)
753
Fig. 4. elongated, suggesting more usual Magnification, X430 ; reduced ‘$6.) to be purely nutritional. (Hema-
DISCUSSIOh‘
Microscopically, these tumors have been divided into a vascular and a nonvascular type. The vascular type has been described as containing numerous thick-walled vessels with small rounded or stellate lumina. Although some cases of subcutaneous leiomyomas have definitely been shown to arise from the walls of veins, there has been some controversy in most cases as to whether the vessels are arteries or veins. Stout” observed that the vessels in these cases were atypical and not identifiable as either arteries or veins but bore more resemblance to veins. We would like to point out that the vascular types in t,he oral cavity have been thought to be derived from the smooth muscle of blood vessels. The nonvascular type is not without blood vessels but contains only a few, which are purely nutritional. Recently Stout reported a group of unusual smooth muscle tumors that are of relatively common occurrence in the stomach. He stated that, because of their bizarre microscopic appearance, these tumors have puzzled the majority of the pathologists and that it is not suprising that they have been incorrectly neurofibromas, neurilemmomas, glomus diagnosed as fibromas, fibrosarcomas, and myriad other entities. He described tumors, granular-cell myoblastomas, the lesions as being comprised predominately of rounded or polygonal cells,
only a. few of whicah c~ontaincd (~longatctl l)lunt-cqrded nuclei that co~rltl 1)~ idcntificd as nl~cl~i ot’ I(~iorrr~o})lwsts. Thch rounded cells cxhibitc,d a nliltll~~ acidophilic cytoplasm, tllv al~scmc ol’ Irlyofihrils, and oftw a ~lcar zone partI!or completely surrounding the nuclt7w. (‘linically and grossly~ these lesions were not unlike the leiomyomas that OWUI’ in other locations. dlthough thr qc>at majority wcw c~omplt~tIely bc&gn in their behavior, two out of the sixty-nincb cases reported proved their malignancy by mrtastasizing. Stout attempted to establish criteria for determining their behavior. The most significant findin g was thcl high mitotic rate in isolated zones of thr two tumors that met~wstasizcd. There wvc~rc nineteen rnitoses for one case ant1 thirteen for the other in fifty high-power fields, which was very high when compared to the remaining sixty-seven tumors. Stout stated that, the only other locations in which he had observed these unusual smooth rnusclc tumors were the intestinal tract and the uterus. Because of the bizarre appcarancc and the rnalignant behavior of a few of these tumors, Stout) oxpressed his opposition to calling them leiomyomas and suggested the term hizcrrw leiomyoblostomcl.’ SUMMARY
AKD
COSCLUSION
We have prtrscnt,ed a case of leiomyoma of the lip and a review of the lit’crature. Onlv eight cases involving the oral cavity hare been reported up to this date, and all have been rc1ativel.v uniform in their clinical and microscopic characteristics. They were all observed to contain fairly well-differentiated smoot,h muscle cells. The case prcscnted here is similar in its clinical features to the previously reported cases but, differs in its microscopic apprarante. Stout agrees that this tumor resembles a group of unusual smooth rriusclc tumors which he has rccc>ntly observed in the stomach. Because most of thcsc tumors were benign, although a small percentage showed malignant behavior, Stout considered it wrong to call them either leiomyomas or leiornyosarcomas. He suggested the terrn leiomyoblastoma, as it denotes neither benignancy nor malignancy. As mentioned previously, the tumors t,hat, showed rnalignant bchavior differed from the benign ones only in the number of mitoses. In this present ease Stout was able to find only two rnitoses in fiftv high-power fields, which he said was a good indication that the tumor was benign.s Of COU~SC, WC cannot be sure of the origin of this smooth muscle tumor, but two possible SOUTCW are (1) the wall of a blood vessel and (2) displaced rmbryonal tissue. The fact that this lesion is not of the vascular type, does not preclude its origin from a blood ~sscl wall. REFERESCES
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E.:
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Volume 17 Number6 G. 7. 8. 9.
LEIOMYOMA
OF
ORAL
CAVITY
755
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