Smooth muscle tumours of the mouth

SMOOTH M U S C L E T U M O U R S OF THE MOUTH D. G. MAcDoNanD, B.D.S. University of Glasgow Dental School INTRODUCTION SMOOTH muscle tumours of the oral cavity are uncommon. Hagy et aL (1964), in a review of the literature, discuss eight previously published cases and present one case of their own. Many current standard textbooks and previously published papers make no mention of leiomyomas occurring in the lips and cheeks. Such tumours are noted in the literature on cutaneous and subcutaneous leiomyomas, and although frequently no details are given about involvement of the oral cavity, these lesions may present a diagnostic problem to the oral surgeon. Origin o f S m o o t h Muscle Tumours. Little smooth muscle is normally present in the oral cavity proper. The walls of blood vessels form the largest single site. Other possible sources are circumvallate papillae (Stout, I938) or heterotopic smooth muscle. The suggestion of Utz (I965) that some tumours may be derived from muscle differentiated from mesoderm in a post-embryonic period or from metastases of uterine myomata seems an unnecessary complication. Duhig and Ayer (I959) suggest a progression of lesions--haemangioma, angioma with much non-striated muscle, vascular leiomyoma, leiomyoma with many vessels and solid leiomyoma--and state that vascular leiomyomas may represent only one stage in a continuous process of smooth muscle proliferation, and an appreciable number may not be true tumours but merely vascular malformations. If this suggestion of a progression of lesions is acceptable, then all the leiomyomas of the oral cavity proper, but not necessarily of the lips and cheeks, may be of vascular origin. Leiomyomas in the skin may be solitary or multiple. As the multiple ones are generally small and superficial they are unlikely to present any problem to the oral surgeon. However, the solitary type is most frequently subcutaneous and may involve the mouth. The sites of origin of these tumours are the arrectores pilorum muscles, the muscle coats of vessels and smooth muscle in the areola and genital regions. Solitary tumours of arrectores pilorum muscle are less common than the multiple superficial tumours mentioned previously, but those derived from the other sites are generally solitary. Stout (I937) quotes the musculi curls diagonales described as a normal structure in the cheek by Hoepke (i927) as the probable source of some tumours, and he illustrates a section of cheek with muscle bundles deep in the corium. Abulafia and Grinspan (1956) do not regard the musculi cutis diagonales as an entity, but think them aberrant hair follicle muscles. Vascular leiomyomas are generally stated to be derived from the muscle coats of veins, but the vessels within the tumours are often thick walled, although they contain no elastic tissue, and frequently have a characteristic stellate lumen. Jansen (1952) demonstrated a valve in a tumour vessel and concluded that they 1 Present address, Department of Oral Medicine, Glasgow Dental Hospital, Glasgow, C3. 207

208

BRITISH JOURNAL OF ORAL SURGERY

were veins. Some authors suggest a derivation from arteriovenous anastomoses (Ekestr6m, r95o ) and point out the similarities to glomus tumours. Review o f the Literature. The cases of leiomyoma previously recorded in the literature are summarised in Table I. TABLE I P R E V I O U S L Y R E C O R D E D L E I O M Y O M A S OF T H E M O U T H Author Blanc (I884) Herzog (I898) Fein (I9o5) Glas (I9o5) *Sehrt (I9O7) Weil (I914) Peter (I937) Stout (I938), Case I Stout (I938), Case 2 Burford et al. (I944) *Ekestr6m (I95o) Abulafia and Grinspan (I956) *Duhig and Ayer (I959) , *Yannopoulos and Stout (I9"62) Yannopoulos and Stout (x962) Kist and Bhaskar (I964) Lucas (I964) Hagy et al. (I964) Bertelli (I965) Utz (I965) Frenkel (I965) • Merrill and Downs (1967) Merrill and Downs (i967)

Sex

Age

M M F M M M F M F M

33 45-50 26 44 Student 69 40 50 29 32

F

48 II

F M M F M F M

4~

58 I2

76 43 29 I3 76 72

Site Tongue " ----i Cheek Palate Tongue Cheek Uvula Floor of Mouth Tongue Tongue Tongue 8 cases in Cheek and External Ear Cheek 2 cases in Lower Lip Cheek Tongue Palate Gingiva Lip Tongue Gingiva Cheek Cheek Tongue

* Full details not given.

Within the mouth, the tongue is the most frequent site, eight cases being recorded. Bertelli (I965) recorded a leiomyoma on the ventral aspect of the tip of the tongue with a maximum diameter of 2"5 cm. The more usual site, however, is the base of the tongue. The largest tumour was 5"5 × 6 × I cm. recorded by Burford et al. (I944). This recurred two months after excision. Merrill and Downs (~967) reported a pedunculated leiomyoma of the tongue in a man of 72 which had been present since birth. A tumour of the floor of the mouth was recorded by Peter (I937). This appeared to arise in relation to the posterior pole of the sublingual salivary gland and prevented the wearing of a lower denture. One case of a small leiomyoma of hard palate was recorded by Kist and Bhaskar (I964), and cases at the posterior border of palate and uvula respectively were noted by Fein (I9o5) and Weil (i914). This latter case was the size of half-

SMOOTH MUSCLE TUMOURS OF THE MOUTH

209

a plum on the posterior border o f the uvula in a man o f 69 who had previously had a leiomyoma o f the larynx. Lucas (I964) illustrated a case which presented as a purple swelling of the gum I cm. in diameter in a boy of I2. Utz (I965) recorded a case in the gingiva of a woman o f 29, four weeks post-partum. This was a pedunculated lesion which increased steadily in size after the extraction of the lower third molar, which was originally believed to be the cause of the swelling. T w o cases of vascular leiomyomas in the lower lip were recorded b y Duhig and Ayer (I959), but full details o f these were not given. Hagy et a l . (I964) described a smooth muscle tumour in the lip o f a 76-year-old woman. I t was not stated whether this was in the upper or lower lip, but the clinical appearances suggested a mucocele. TABLE II PREVIOUSLY RECORDED LEIOMYOSARCOMAS OF THE M O U T H Author Zieler (I9O8)i Hayn (I9IO) ] *Diss and L6vy (1924) • *Milian et al. (1926, 1927) • Ackerman and Stout (1949)/ Stout and Hill (I958) j Yannopoulos and Stout (1962) Miles and Waterhouse (r962) Pettini (r965)

Sex

Age

F F

67

F M

61

M F

II

months 34 73

Site Cheek Cheek Cheek Cheek Tongue Mandible Cheek

* Full details not given. Details o f cases in the cheek are more difficult to review because they are noted in a more varied range of publications. Ekestr6m (195o) recorded eight cases of angioleiomyoma in the cheeks and external ear, but did not give further details o f these. H e noted, however, that one was painful. Herzog (I898) reported a painful tumour in the cheek i cm. in diameter and Abulafia and Grinspan (I956) reported a t u m o u r 2 cm. in diameter which was painful on palpation and very hard. T h e case in.the cheek recorded b y Sehrt (19o7) recurred twice after excision and was painful on pressure. T h e primary tumour and recurrences were each approximately 2"75 cm. long by o'75 cm. in diameter and were all related to blood vessels. T w o cases in the cheek are recorded in children (Yannopoulos & Stout, I962; Frenkel, I965). Frenkel's case in a boy of 13 caused slight difficulty in opening the mouth, but was painless. T h e turnout was 3 × 2 × I cm. and was regarded as a leiomyofibroangioma (glomangioma) derived from an arterio-venous anastomosis. A 76-year-old man with two firm movable subcutaneous and submucosal leiomyomas in the cheek was noted by Merrill and Downs (I967). Seven cases o f leiomyosarcoma of the m o u t h are noted in the literature (Table II).

210

BRITISH

JOURNAL

OF ORAL S U R G E R Y

Zieler (19o8) and H a y n (191o) both discussed the same case in the cheek. T h e two cases reported b y Diss and Ldvy (I924), Milian and P&in (1926) and Milian et aL (1927) both occurred in the cheek in old lupus vulgaris scars. Ackerman and Stout (1949) reported a leiomyosarcoma in the cheek of a woman o f 61. T h i s had been present for about six months and was easily enucleated. Stout and Hill (1958) noted that this woman died six years later with local recurrence and pulmonary metastases. Pettini's (1965) case was also in the cheek o f an elderly woman. T h e t u m o u r was the size o f an egg and the oral mucosa covering it was stretched. T h e case recorded by Yannopoulos and Stout (1962) was a polypoid turnout I "5 cm. in diameter in the tip o f the tongue of a male child. Miles and Waterhouse (1962) reported a leiomyosarcoma in a man of 34 which caused pain and swelling in the region of the lower second and t h i r d molars and involved the bone of the mandible. Secondary t u m o u r was present in submandibular lymph nodes. T r e a t m e n t by surgery and radiotherapy was followed by local recurrence and lung and bone metastases resulting in death within two years. CASE R E P O R T S Case I. A 44-year-old male presented with a hard swelling I "5 × I cm. in the upper lip at the corner of the mouth, more obvious from the mucous membrane aspect. It

FIG. I

Case I. Leiomyoma; note numerous thick-waUed vessels. H. & V.G.

× I2O.

had been present for two years, but was not painful. It shelled out easily from an incision in the mucous membrane and the subsequent healing was uneventful. Pathology. Naked Eye. Firm white nodule I cm. in diameter. Histology. Reveals a leiomyoma which is encapsulated and consists of irregular columns and cords of smooth muscle cells with numerous vascular channels (Fig. I).

SMOOTH MUSCLE TUMOURS OF THE MOUTH

211

C a s e 2. A n I I - y e a r - o l d schoolgirl p r e s e n t e d w i t h an apparently cystic swelling on the d o r s u m of t h e tongue; a history o f a m i n o r b u r n to t h e t o n g u e p r e c e d e d the appearance o f the turnout.

FIG. 2 Case 2. N o t e m o r e solid area of smooth muscle and adjacent areas of myxomatous degeneration. H. & E. × 13.

Case 2.

FIG. 3 S m o o t h muscle in m o r e solid area o f turnout. H.&E. ×IIO.

212

BRITISH JOURNAL

OF ORAL SURGERY

The lesion was excised under local anaesthesia with an ellipse of overlying mucosa. Pathology. Naked Eye. Ellipse of mucosa 1.5 × I cm. with central cystic swelling o.5 cm. in diameter. Histology. The lesion is a leiomyoma which is encapsulated and shows some areas of myxomatous degeneration. The more solid parts of the tumour are notably vascular and the lesion is probably derived from vascular smooth muscle (Figs. 2 and 3). Careful investigation in both cases reveals no pallisading and the haematoxylin and eosin preparations show the typical appearances of smooth muscle. Special stains show the usual rinctorial reactions of smooth muscle, but myofibrils are not demonstrated. Silver reticulin stains show the characteristic fine reticulin pattern around individual muscle cells. Using the criteria of Duhig and Ayer (1959) that the vascular component is greatly in excess of that required for nutrition, these tumours are both classified as vascular leiomyomas. DISCUSSION There is a paucity of smooth muscle in the oral cavity, the largest single site being in vessel walls, and the majority of smooth muscle tumours in the mouth are probably derived from this source. Of the nine recorded cases of leiomyoma in the tongue, four were thought to be of vascular origin. Stout (I938) regarded his Case 2 and those of Blanc (I884) and Glas (r9o5) as possibly derived from circumvallate papillae, although Glas suggested the thyroglossal duct as the site of origin. The site of origin of the remaining two cases in the tongue is not stated, but they were probably vascular. The illustrations in Peter's (I937) case in the floor of the mouth show that it was possibly a vascular leiomyoma although this is not stated. The presence of a capsule was noted in three of these tumours and myxomatous degeneration was present in one, which also had a history of minor trauma preceding the tumour. The turnouts in the palate (Kist & Bhaskar, 1964) and in the uvula (Weil, I9r4) were of vascular origin while Fein's (i9o5) case of a pedunculated lesion at the posterior border of the palate was regarded as a tumour of heterotopic smooth muscle. None of these three cases was encapsulated. The cases in the gingiva described by Lucas (I964) and by Utz (I965) were possibly of vascular origin, and the latter was encapsulated. Of these intra-oral turnouts only one was recorded as painful, and this was due to ulceration of the overlying epithelium. This was the case of Burford et aL (I944) which was the largest and also the only one to recur. Four cases of leiomyomas are noted in the lips. Of these, three were vascular leiomyomas and the fourth was probably vascular in origin. Eleven cases of vascular leiomyomas in the cheek are recorded. The site of origin of the case of Yannopoulos and Stout (I962) was not noted. Frenkel (I965) and Ekestr6m (I95o) suggested a derivation from arterio-venous anastomoses. Vascular leiomyomas in the lips and cheeks are generally encapsulated. Two cases of solitary leiomyomas in the cheek derived from hair follicle muscle (Herzog, I898; Abulafia & Grinspan, I956) were not encapsulated. Three of these tumours in the lips and cheeks were painful and one was noted as recurring twice after excision. In one case (Merrill & Downs, I967) two leiomyomas were present. The age range of these leiomyomas of the mouth varies between 4½ years and 76 years and tumours are recorded in males slightly more frequently than in females.

SMOOTH MUSCLE TUMOURS OF THE MOUTH

213

Malignant tumours of smooth muscle are recorded in the cheeks, tongue and mandible. Determination of malignancy was mainly on the cellular picture; high mitotic rate, nuclear pleomorphism and hyperchromatism and bizarre cell forms being taken as indicative of malignancy. This picture may only be present in parts of the tumour and complete examination of the material is important. The case of Miles and Waterhouse (1962) did not show frequent mitotic figures, but invaded locally and metastasised to lymph nodes, lung and bone. Stout and Hill's (1958 ) case also died with local recurrence and pulmonary metastases. Extensive local spread was noted by Diss and L6vy (1924) and Hayn (191o) recorded both local and blood vessel invasion. The two cases recorded by Diss and L6vy (1924) and Milian and P6rin (1926) and Milian et al. (1927) both occurred in old lupus vulgaris scars, but as the reports are brief and no illustrations are available, interpretation is difficult. Cases of leiomyosarcoma in the cheek where the sex was recorded were all in women, and in the three cases where the age was noted, this was over 6o. Yannopoulos and Stout's (1962) case is noteworthy in being the only case in the tongue and also in a child. The child was well, with no evidence of a recurrence, four years and eight months after excision of this turnout. It would appear that smooth muscle turnouts of the mouth are probably not so uncommon as is generally supposed and malignant variants do occur. There are no reliable clinical diagnostic features of these tumours and the diagnosis must be made histologically. SUMMARY The literature on smooth muscle tumours of the mouth, including those of lips and cheeks, is reviewed and two new cases are recorded. The presence of malignant variants is described and the importance of adequate histological exam nation is noted. ACKNOWLED GEMENTS I wish to thank Professor T. Symington and Dr. H. W. Noble for their encouragement and assistance and Dr. D. A. Lunt for assistance with translation of original papers.

REFERENCES ABULAFIA,J. & GRINSPAN, D. (1956). Archos argent. Derm. 6, I. ACKERMAN, L. V. & STOUT, A. P. (1949). Cancer Seminar. I, 15. BERTELLI, A. DE P. (1965). Oral Surg. I9, 771. BLANC, E. (1884). Gaz. hebd. Mdd. Chir. 21 (2 *'m~ Set.), 611. BURFORD, W. M., ACKERMAN,L. V. & ROBINSON, H. B. G. (1944). Am. ft. Orthod. 3o, 395. DISS, A. & L~vY, G. (1924). Bull. Soc. ft. Derm. Syph. 31, lO8. DUHIG, J. T. & AYER, L. P. (1959). Archs Path. 68, 424. EKESTROM, S. (195o). Acta path. microbiol, scand. 27, 86. FEIN, J. (19o5). Arch. Lar. Rhinol. 17, 533. FRENKEL, G. (1965). Dr. zahmirztl. Z. 2o, 168. GLAS, E. (19o5). Wien. hlin. Wschr. 18, 746. HAGY, D. M., HALPERIN, V. & WOOD, C. (1964). Oral Surg. 17, 748. HAYN, F. (191o). Arch. Derm. Syph. lO5, 211. HERZOG, i . (1898). ft. cutan, genito-urin. Dis. 16, 527 .

214

BRITISH JOURNAL OF ORAL SURGERY

HOEPKE, H. (1927). Die Haut. I n Mollendorff's Handbuch der Mikroskopischen Anatomy des Menschen, Berlin, I I I/I, 39JANSEN, L. H. (1952). Acta derm-venerol. 32, 40. KIST, R. & BHASKAR,S. (1964). J. oral Surg. 22, 346. LUCAS, R. B. (1964). Pathology of Tumours of the Oral Tissues, p. 17o. London: Churchill. MERRILL, R. G. & DOWNS, J. R. (1967). Oral Surg. 23, 438. MILES, A. E. W. & WATERHOUSE,J. P. (1962). J. Path. Bact. 83, 551. MILIAN, G. & PI~RIN,L. (1926). Bull. Soc. fr. Derm. Syph. 33, 621. MILIAN, G., PI~RIN, L. & DELARUE(1927). Bull. Soc. ft. Derm. Syph. 34, 432. PETER, K. (1937). Dr. Zahn- Mund- u. Kieferheilk, 4, 412. PETTINI, P. (1965). Annali Stomat. 14, 965. SEHRT, E. (19o7). Beitr. klin. Chir. 54, 732. STOUT, A. P. (1937). Am. J. Cancer, 2% 435. STOUT, A. P. (1938). Am. ]. Cancer, 34, 31. STOUT, A. P. & HILL, W. T. (1958). Cancer, I I , 844. UTZ, W. (1965). Stoma, 18, 19o. WEIL, I. (1914). Mschr. Ohrenheilk. Lar.-Rhinol. 48, lOO2. YANNOI'OULOS,K. & STOUT, A. P. (1962). Cancer, I3~ 958. ZIELER (I908). Verh. dr. path. Ges. 12, 142.