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Leiomyoma of the tongue presenting as congenital airway obstruction
INTEBNATlOWJOUWALOT
Adatric
ELSEVIER SCIENCE IRELAND
International Journal of Pediatric Otorhinolaryngology 29 (1994) 139-145
Leiomyoma of the tongue presenting as congenital airway obstruction Howard S. Kotlera, Nevenka S. Gouldb, Benjamin Gruber*a ‘Departmem of Otolaryngology. Head and Neck Surgery, University of Illinois College of Medicine, Chicago, IL, USA ‘Department of Pathology, Michael Reese Hospital and Medical Center, Chicago. IL, USA
(Received I I February 1993; revision received 7 September 1993; accepted 12 September 1993)
Abstract Leiomyoma is a benign tumor of smooth muscle origin that is most commonly found in the uterus, gastrointestinal tract, and skin. Intraoral leiomyomas are rare, usually asymptomatic, and, most often present in the fifth decade of life. A review of the literature since 1884 yielded only 125 cases, including 23 of the tongue. We report the first case of a congenital leiomyoma of the tongue presenting with airway obstruction. A 6-day-old female infant, intubated since birth for respiratory distress, was evaluated for an obstructing oropharyngeal mass. The tumor was pedunculated, mucosa-covered, and localized to the posterior tongue. The mass was completely excised using a CO2 laser. There has been no recurrence in 1 year of follow-up.
Key words: Leiomyoma; Neoplasm of tongue; Airway obstruction;
Infants
1. Introduction
Leiomyoma is a benign neoplasm of smooth muscle origin that most frequently occurs in the uterine myometrium [19]. The gastrointestinal tract, skin and subcutaneous tissues are less frequent sites [26], and occurrence within the oral cavity is considered rare, probably because of the paucity of smooth muscle tissue. Besides * Corresponding author, Department of Otolaryngology, Head and Neck Surgery, Michael Reese Hospital and Medical Center, 2929 South Ellis Avenue, Chicago, IL 60616, USA. Presented at the 20th annual meeting of the Society for Ear, Nose & Throat Advances in Children, Toronto, Canada, December 1992. 0165-5876/94/$07.00 0 1994 Elsevier Science Ireland Ltd. All rights reserved. SSDI 0165-5876(93)00985-N
140
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol,
29 (1994)
139-145
the arterial tunica media suggested by Stout [21], several sources of smooth muscle within the oral cavity of smooth muscle have been described [4,10,24]. Glas, in 1905, suggested the smooth muscle of the ductus lingualis, and proposed the circumvallate papillae as the origin of the tumor [l 11. Since the first reported case of an oral leiomyoma by Blanc in 1884 [2], over 125 cases, including 23 of the tongue, have been reported in the world literature [ 131. While most oral leiomyomas are usually asymptomatic, tongue lesions may present as painful masses, a progressive change in voice, persistent sore throat or trismus [l-3,9,18,21]. We report the first case of a congenital leiomyoma of the tongue presenting as airway obstruction. 2. Case report A 6-day-old black female, intubated since birth, was evaluated by the Otolaryngology, Head and Neck Surgery service for airway obstruction and a pharyngeal mass. The patient was born at 38 weeks gestation with Apgar scores of 6 and 8, but was noted immediately postpartum to have marked substernal retractions, nasal flaring, and cyanosis. Examination of the airway revealed a pedunculated, mucosa-covered soft-tissue tongue mass. The mass was situated in the midline, approximately 5 mm anterior to the circumvallate papillae, retrodisplaced the soft palate and presumably was responsible for the upper airway obstruction (Fig. 1). Following nasotracheal intubation, the patient remained stable.
Fig. 1. Clinical photographs of tongue tumor in this neonatal patient.
H.S. Kotler et al. /Int. J. Pediatr.
Fig. 2. Technicium
thyroid
Otorhinolaryngol.
scan demonstrating
29 (1994)
normal
uptake
139-145
141
in neck and no significant
uptake
in the
oral cavity.
A technicium-99 thyroid scan was performed to exclude the possibility of ectopic thyroid tissue (see Fig. 2). The scan showed normal Tc uptake in the neck, but no active thyroid tissue in the tongue. The remainder of physical examination, chest radiograph and laboratory studies were all within normal limits. The patient underwent laryngoscopy and CO* laser excision of the mass. She recovered uneventfully, and had a normal oral intake by postoperative day 2. She was discharged on postoperative day 5, and has remained clinically free of disease 6 months after excision. The specimen consisted of a tan, mucosa-covered elastic piece of tissue measuring approximately 1.5 cm. x 1.0 cm. x 0.5 cm (Fig. 3). Histologically, the mass con-
Fig. 3. Excised lingual
leiomyoma
from 6-day-old
girl.
142
Fig. 4. Photomicrograph
H.S. Korler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994)
139-145
demonstrating typical appearance of benign leiomyoma. (H & E, x SO.)
sisted primarily of well-differentiated bundles of spindle cells, with elongated uniform blunt-poled nuclei and eosinophilic focally vacuolated cytoplasm. Peripheral to the mass were normal tissue elements including squamous mucosa, minor salivary glands, nerves and fibrovascular structures (Fig. 4). Immuno-peroxidase stains were strongly positive for desmin and smooth muscle actin. The final diagnosis after consultation was congenital leiomyoma of the tongue. 3. Review of literature Since the first reported leiomyoma in 1884, over 125 cases of intraoral leiomyoma have been described [13]. The extensive review of 7748 smooth muscle tumors by Farman in 1975 reported only 0.064% had an intraoral location [S]. The most common intraoral sites are the lips (24.4%), followed by the palate (21.1%) tongue (19.5%) and cheek (16.3%) [2]. A total of 23 cases of leiomyoma localized to the tongue have been described. These tumors are usually asymptomatic and recognized on routine oral examination as a benign space-occupying lesion. However, patients may present with a variety of complaints including change in voice, sore throat, trismus, difficulty chewing and pain at the tumor location [2,3,9]. Clinically, lingual leiomyomas tumors may be firm submucosal or exophytic nonulcerated masses. Varying from a few mm up to 3 cm, the tumor may be pedunculated or sessile, and covered with normal appearing mucosa. There is no site specificity, and they may be found on the antero- or postero-lateral aspect, dorsal surface, adjacent to the foramen cecum, or overlying the circumvallate papillae. Watabe and Morishima in 1963 reported a case at the root of an infant’s tongue [25], and in 1967 Merill and Downs cited a case of median glossal leiomyoma in a 72-yearold [15].
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994)
139-145
143
There are few reported cases of pediatric lingual leiomyoma in the world literature. Yannopoulos and Stout described eleven cases of oral leiomyoma in children, including one on the tongue of a 4-year-old girl which was treated by complete excision [26]. MacDonald, in 1969, reported on an 1l-year-old female with a cystic swelling on the dorsum of the tongue after a history of thermal trauma [14]. The mass was excised with an ellipse of overlying mucosa, and histologically diagnosed as a vascular leiomyoma. Farman briefly cited, without description of location or treatment, a case of an I l-year-old female with a lingual leiomyoma [7]. No details of recurrence or duration of follow-up are given in any of these three cases. Haedicke and Kaban, in 1988, reported an S-year-old boy who presented with a 6-month history of intermittent pharyngeal pain [ 121.A leiomyoma was excised from the left lateral border of the tongue with l-cm margins. Follow-up was only 1 month. According to Enzinger et al., the World Health Organization describes three types of leiomyoma: leiomyoma (solid), angiomyoma (vascular), and epithelioid leiomyoma (leiomyoblastoma) [6]. The solid leiomyoma is not associated with blood vessels and is composed of bunches of intertwining spindle cells with varying amounts of collagen. The collagen may predominate so that the true nature of the lesion may become obscured. Most leiornyomas of the tongue are of this nonvascular type. Unlike solid leiomyomas, vascular leiomyomas are intimately associated with the vascular tunica media from which they may arise. These tortuous tumors are composed of thick-walled vessels, with smooth muscle bundles circularly arranged about a small rounded lumen. Duhig and Ayer have proposed that the vascular leiomyoma is but one type along a continuum of smooth muscle proliferative tumors including hemangioma, angioma, vascular leiomyoma, and solid leiomyoma [5]. The leiomyoblastoma is composed of rounded or polygonal cells with acidophilic cytoplasm and clear perinuclear space. Smooth muscle fibers are rarely found. Svane et al. found that 73% of oral leiomyomas were angiomyomas, 23% were solid leiomyomas and that leiomyoblastoma accounted for less than 1% [23]. The age range for all intraoral leiomyomas is 2 months to 83 years, with the greatest incidence between 40 and 59 years. The age distribution for leiomyoma of the tongue appears bimodal, but the limited number of cases does not allow for any real pattern recognition. While intrauterine leiomyomas are estrogen dependent, developing in 25% of women during their active reproductive life, there is no conclusive evidence that any sex predilection exists for oral leiomyoma [19]. A 1:1 sex ratio was reported by Leung et al. for 125 cases of oral leiomyoma [ 131. However, Praal et al. concluded that there is a predilection of oral leiomyoma for women under 30 years of age and for men older than 30 [18]. The diagnosis of oral leiomyoma is based primarily on histologic grounds, as these tumors may clinically resemble leiomyosarcoma, fibroma, neurofibroma or lipoma. Microscopic identification may prove difficult in differentiating leiomyoma from neurofibroma, neurilemmoma, spindle cell pleomorphic adenoma or welldifferentiated leiomyosarcoma. Histological differentiation between leiomyoma and malignant leiomyosarcoma is particularly important, because oral smooth muscle tumors have a higher propensity toward malignancy when compared with extraoral leiomyomas. According to Farman and Kay, 20% of the oral smooth muscle tumors reported in the literature were malignant [8]. Robbins et al. state that ten mitotic
144
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994) 139-145
figures per ten high power field indicates probable malignancy and a tumor with fewer than two mitoses per ten high power field carries a favorable prognosis [19]. Although the number of mitoses is considered by some investigators the most important indicator in differentiating leiomyosarcoma from its benign counter-part, this criterion cannot be used reliably. Histologically benign smooth muscle tumors have been reported to be locally invasive or metastatic to regional lymph nodes or bones [16,22]. 4. Treatment Complete surgical excision has been advocated as the primary treatment modality, given the high incidence of malignancy. Leiomyoma of the tongue, especially those located submucosally may not have a well-defined capsule, making the possibility of recurrence more likely if generous margins are not included in the specimen. Thus far, four cases of recurrence have been reported, after variable follow-up periods. Recurrence is rarely seen after 6 years [3,17,20,21]. Each of these recurrent leiomyoma were histologically confirmed as the vascular type (angiomyoma). Local excision with a l-cm margin is the treatment of choice, as radiotherapy and chemotherapy have no proven efficacy. Due to the age and size of our patient, such extensive resection was not possible. Gross margins of 0.5 cm were obtained, and to date there has been no recurrence. 5. References Bertelli, A. (1965) Uncommon tumors of the tongue (lipoma and leiomyotibroma): report of two cases. Oral Surg. 19, 771-775. Blanc, E. (1884) Contribuition a l’etude des tumeurs fibreuses de la langue: fibromyome de la langue. Gaz. Hebd. Med. 21, 611-613. Burford, W.N., Ackerman, L.V. and Robinson, H.B.G. (1944) Leiomyoma of the tongue. Am. J. Orthod. (Oral Surg.) 30, 395-397 Cherrick, H.M., Dunlap, C.L. and King, O.H. (1973) Leiomyomas of the oral cavity: review of the literature and clinicopathologic study of seven new cases. Oral Surg. 35, 54-66. Duhig, J.T. and Ayer, J.P. (1959) Vascular leiomyoma: a study of sixty-one cases. Arch. Pathol. 68, 424-430. Enzinger, F.M., Lattes, R. and Torloni, H. (1969) Histologic Typing of Soft Tissue Tumours. World Health Organization, Geneva, pp. 30-31. Farman, A.G. (1975) Benign smooth muscle tumors. S. Afr. Med. J. 49, 1333-1340 Farman, A.G. and Kay, S. (1977) Oral leiomyosarcoma: report of a case and review of the literature pertaining to smooth muscle tumors of the oral cavity. Oral Surg. 43, 402-409. 9 Erenkel, G. (1965) Auftreten eines Leiomyofibroangioms im Wangen- und Jochbogenbereich. Dtsch Zahnaerztl Z. 20, 168-172. 10 Garrett, J.R. (1969) Angiomyoma of the palate: report of a case. Oral Surg. 27, 103-105. 11 Glas, E. (1905) Beitrage zur Pathologie der Zungengrundtumoren. Wien. Klin. Wochenschr. 18, 746-752. 12 Haedicke, G. and Kaban, L. (1988) Smooth-muscle tumors of the oral cavity. Plast. Reconstr. Surg. 81, 264-269 13 Leung, K.W., Wong, D.Y.K. and Li, W.Y. (1990) Oral leiomyoma: case report. J. Oral Maxillofac. Surg. 48, 735-738. 14 MacDonald, D.G. (1969) Smooth muscle tumors of the mouth. Br. J. Oral Surg. 6, 204-214 15 Merrill, R.G. and Downs, J.R. (1967) Oral leiomyomas: report of two cases. Oral Surg. 23,438-442.
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994) 16
17 18 19 20 21 22 23 24 25 26
139-145
145
Miles, A.E.W. and Waterhouse, J.P. (1962) A leiomyosarcoma of the oral cavity with metastasis to lymph-glands. J. Pathol. Bact. 83, 551-555. Natiella, J.R., Neiders, M.E. and Greene, G.W. (1982) Oral leiomyoma: report of six cases and a review of the literature. J. Oral Pathol. II, 353-365. Praal, F.R., Ioannides, CA., van Beek, G.J. and van der Molengraft, F. (1982) Oral leiomyomas. J. Maxillofac. Surg. 10, 229-235. Robbins, S.L., Cotran, R.S. and Kumar, V. (1984) Pathologic Basis of Disease, 3rd Edn. W.B. Saunders Co., Philadelphia, p. 271. Sehrt, E. (1907) Subcutane Leiomyome der Wange und ihre Histologenese. Bruns’ Beitr. Klin. Chir. 54, 723-734 Stout, A.P. (1938) Leiomyoma of the oral cavity. Am. J. Cancer 34, 31-36. Stout, A.P. and Hill, W.T. (1958) Leiomyosarcoma of the superficial soft tissues. Cancer I I, 844-854. Svane, T.J., Smith, B.R., Cosentino, B.J. et al. (1986) Oral leiomyomas: review of the literature and report of a case of palatal angioleiomyoma. J. Peridontol. 57, 433-430. Utz, W. (1965) Beitriige zur Klinik und Pathologic des Leiomyomas in der Mundhiihle. Stoma 18, 190-192. Watabe, T. and Morishima, T. (1963) Rare case of leiomyoma at the root of the tongue in an infant. Otolaryngology (Tokyo) 35, 147-148. Yannopoulos, K. and Stout, A.P. (1962) Smooth muscle tumors in children. Cancer 15, 958-971.
Adatric
ELSEVIER SCIENCE IRELAND
International Journal of Pediatric Otorhinolaryngology 29 (1994) 139-145
Leiomyoma of the tongue presenting as congenital airway obstruction Howard S. Kotlera, Nevenka S. Gouldb, Benjamin Gruber*a ‘Departmem of Otolaryngology. Head and Neck Surgery, University of Illinois College of Medicine, Chicago, IL, USA ‘Department of Pathology, Michael Reese Hospital and Medical Center, Chicago. IL, USA
(Received I I February 1993; revision received 7 September 1993; accepted 12 September 1993)
Abstract Leiomyoma is a benign tumor of smooth muscle origin that is most commonly found in the uterus, gastrointestinal tract, and skin. Intraoral leiomyomas are rare, usually asymptomatic, and, most often present in the fifth decade of life. A review of the literature since 1884 yielded only 125 cases, including 23 of the tongue. We report the first case of a congenital leiomyoma of the tongue presenting with airway obstruction. A 6-day-old female infant, intubated since birth for respiratory distress, was evaluated for an obstructing oropharyngeal mass. The tumor was pedunculated, mucosa-covered, and localized to the posterior tongue. The mass was completely excised using a CO2 laser. There has been no recurrence in 1 year of follow-up.
Key words: Leiomyoma; Neoplasm of tongue; Airway obstruction;
Infants
1. Introduction
Leiomyoma is a benign neoplasm of smooth muscle origin that most frequently occurs in the uterine myometrium [19]. The gastrointestinal tract, skin and subcutaneous tissues are less frequent sites [26], and occurrence within the oral cavity is considered rare, probably because of the paucity of smooth muscle tissue. Besides * Corresponding author, Department of Otolaryngology, Head and Neck Surgery, Michael Reese Hospital and Medical Center, 2929 South Ellis Avenue, Chicago, IL 60616, USA. Presented at the 20th annual meeting of the Society for Ear, Nose & Throat Advances in Children, Toronto, Canada, December 1992. 0165-5876/94/$07.00 0 1994 Elsevier Science Ireland Ltd. All rights reserved. SSDI 0165-5876(93)00985-N
140
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol,
29 (1994)
139-145
the arterial tunica media suggested by Stout [21], several sources of smooth muscle within the oral cavity of smooth muscle have been described [4,10,24]. Glas, in 1905, suggested the smooth muscle of the ductus lingualis, and proposed the circumvallate papillae as the origin of the tumor [l 11. Since the first reported case of an oral leiomyoma by Blanc in 1884 [2], over 125 cases, including 23 of the tongue, have been reported in the world literature [ 131. While most oral leiomyomas are usually asymptomatic, tongue lesions may present as painful masses, a progressive change in voice, persistent sore throat or trismus [l-3,9,18,21]. We report the first case of a congenital leiomyoma of the tongue presenting as airway obstruction. 2. Case report A 6-day-old black female, intubated since birth, was evaluated by the Otolaryngology, Head and Neck Surgery service for airway obstruction and a pharyngeal mass. The patient was born at 38 weeks gestation with Apgar scores of 6 and 8, but was noted immediately postpartum to have marked substernal retractions, nasal flaring, and cyanosis. Examination of the airway revealed a pedunculated, mucosa-covered soft-tissue tongue mass. The mass was situated in the midline, approximately 5 mm anterior to the circumvallate papillae, retrodisplaced the soft palate and presumably was responsible for the upper airway obstruction (Fig. 1). Following nasotracheal intubation, the patient remained stable.
Fig. 1. Clinical photographs of tongue tumor in this neonatal patient.
H.S. Kotler et al. /Int. J. Pediatr.
Fig. 2. Technicium
thyroid
Otorhinolaryngol.
scan demonstrating
29 (1994)
normal
uptake
139-145
141
in neck and no significant
uptake
in the
oral cavity.
A technicium-99 thyroid scan was performed to exclude the possibility of ectopic thyroid tissue (see Fig. 2). The scan showed normal Tc uptake in the neck, but no active thyroid tissue in the tongue. The remainder of physical examination, chest radiograph and laboratory studies were all within normal limits. The patient underwent laryngoscopy and CO* laser excision of the mass. She recovered uneventfully, and had a normal oral intake by postoperative day 2. She was discharged on postoperative day 5, and has remained clinically free of disease 6 months after excision. The specimen consisted of a tan, mucosa-covered elastic piece of tissue measuring approximately 1.5 cm. x 1.0 cm. x 0.5 cm (Fig. 3). Histologically, the mass con-
Fig. 3. Excised lingual
leiomyoma
from 6-day-old
girl.
142
Fig. 4. Photomicrograph
H.S. Korler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994)
139-145
demonstrating typical appearance of benign leiomyoma. (H & E, x SO.)
sisted primarily of well-differentiated bundles of spindle cells, with elongated uniform blunt-poled nuclei and eosinophilic focally vacuolated cytoplasm. Peripheral to the mass were normal tissue elements including squamous mucosa, minor salivary glands, nerves and fibrovascular structures (Fig. 4). Immuno-peroxidase stains were strongly positive for desmin and smooth muscle actin. The final diagnosis after consultation was congenital leiomyoma of the tongue. 3. Review of literature Since the first reported leiomyoma in 1884, over 125 cases of intraoral leiomyoma have been described [13]. The extensive review of 7748 smooth muscle tumors by Farman in 1975 reported only 0.064% had an intraoral location [S]. The most common intraoral sites are the lips (24.4%), followed by the palate (21.1%) tongue (19.5%) and cheek (16.3%) [2]. A total of 23 cases of leiomyoma localized to the tongue have been described. These tumors are usually asymptomatic and recognized on routine oral examination as a benign space-occupying lesion. However, patients may present with a variety of complaints including change in voice, sore throat, trismus, difficulty chewing and pain at the tumor location [2,3,9]. Clinically, lingual leiomyomas tumors may be firm submucosal or exophytic nonulcerated masses. Varying from a few mm up to 3 cm, the tumor may be pedunculated or sessile, and covered with normal appearing mucosa. There is no site specificity, and they may be found on the antero- or postero-lateral aspect, dorsal surface, adjacent to the foramen cecum, or overlying the circumvallate papillae. Watabe and Morishima in 1963 reported a case at the root of an infant’s tongue [25], and in 1967 Merill and Downs cited a case of median glossal leiomyoma in a 72-yearold [15].
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994)
139-145
143
There are few reported cases of pediatric lingual leiomyoma in the world literature. Yannopoulos and Stout described eleven cases of oral leiomyoma in children, including one on the tongue of a 4-year-old girl which was treated by complete excision [26]. MacDonald, in 1969, reported on an 1l-year-old female with a cystic swelling on the dorsum of the tongue after a history of thermal trauma [14]. The mass was excised with an ellipse of overlying mucosa, and histologically diagnosed as a vascular leiomyoma. Farman briefly cited, without description of location or treatment, a case of an I l-year-old female with a lingual leiomyoma [7]. No details of recurrence or duration of follow-up are given in any of these three cases. Haedicke and Kaban, in 1988, reported an S-year-old boy who presented with a 6-month history of intermittent pharyngeal pain [ 121.A leiomyoma was excised from the left lateral border of the tongue with l-cm margins. Follow-up was only 1 month. According to Enzinger et al., the World Health Organization describes three types of leiomyoma: leiomyoma (solid), angiomyoma (vascular), and epithelioid leiomyoma (leiomyoblastoma) [6]. The solid leiomyoma is not associated with blood vessels and is composed of bunches of intertwining spindle cells with varying amounts of collagen. The collagen may predominate so that the true nature of the lesion may become obscured. Most leiornyomas of the tongue are of this nonvascular type. Unlike solid leiomyomas, vascular leiomyomas are intimately associated with the vascular tunica media from which they may arise. These tortuous tumors are composed of thick-walled vessels, with smooth muscle bundles circularly arranged about a small rounded lumen. Duhig and Ayer have proposed that the vascular leiomyoma is but one type along a continuum of smooth muscle proliferative tumors including hemangioma, angioma, vascular leiomyoma, and solid leiomyoma [5]. The leiomyoblastoma is composed of rounded or polygonal cells with acidophilic cytoplasm and clear perinuclear space. Smooth muscle fibers are rarely found. Svane et al. found that 73% of oral leiomyomas were angiomyomas, 23% were solid leiomyomas and that leiomyoblastoma accounted for less than 1% [23]. The age range for all intraoral leiomyomas is 2 months to 83 years, with the greatest incidence between 40 and 59 years. The age distribution for leiomyoma of the tongue appears bimodal, but the limited number of cases does not allow for any real pattern recognition. While intrauterine leiomyomas are estrogen dependent, developing in 25% of women during their active reproductive life, there is no conclusive evidence that any sex predilection exists for oral leiomyoma [19]. A 1:1 sex ratio was reported by Leung et al. for 125 cases of oral leiomyoma [ 131. However, Praal et al. concluded that there is a predilection of oral leiomyoma for women under 30 years of age and for men older than 30 [18]. The diagnosis of oral leiomyoma is based primarily on histologic grounds, as these tumors may clinically resemble leiomyosarcoma, fibroma, neurofibroma or lipoma. Microscopic identification may prove difficult in differentiating leiomyoma from neurofibroma, neurilemmoma, spindle cell pleomorphic adenoma or welldifferentiated leiomyosarcoma. Histological differentiation between leiomyoma and malignant leiomyosarcoma is particularly important, because oral smooth muscle tumors have a higher propensity toward malignancy when compared with extraoral leiomyomas. According to Farman and Kay, 20% of the oral smooth muscle tumors reported in the literature were malignant [8]. Robbins et al. state that ten mitotic
144
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994) 139-145
figures per ten high power field indicates probable malignancy and a tumor with fewer than two mitoses per ten high power field carries a favorable prognosis [19]. Although the number of mitoses is considered by some investigators the most important indicator in differentiating leiomyosarcoma from its benign counter-part, this criterion cannot be used reliably. Histologically benign smooth muscle tumors have been reported to be locally invasive or metastatic to regional lymph nodes or bones [16,22]. 4. Treatment Complete surgical excision has been advocated as the primary treatment modality, given the high incidence of malignancy. Leiomyoma of the tongue, especially those located submucosally may not have a well-defined capsule, making the possibility of recurrence more likely if generous margins are not included in the specimen. Thus far, four cases of recurrence have been reported, after variable follow-up periods. Recurrence is rarely seen after 6 years [3,17,20,21]. Each of these recurrent leiomyoma were histologically confirmed as the vascular type (angiomyoma). Local excision with a l-cm margin is the treatment of choice, as radiotherapy and chemotherapy have no proven efficacy. Due to the age and size of our patient, such extensive resection was not possible. Gross margins of 0.5 cm were obtained, and to date there has been no recurrence. 5. References Bertelli, A. (1965) Uncommon tumors of the tongue (lipoma and leiomyotibroma): report of two cases. Oral Surg. 19, 771-775. Blanc, E. (1884) Contribuition a l’etude des tumeurs fibreuses de la langue: fibromyome de la langue. Gaz. Hebd. Med. 21, 611-613. Burford, W.N., Ackerman, L.V. and Robinson, H.B.G. (1944) Leiomyoma of the tongue. Am. J. Orthod. (Oral Surg.) 30, 395-397 Cherrick, H.M., Dunlap, C.L. and King, O.H. (1973) Leiomyomas of the oral cavity: review of the literature and clinicopathologic study of seven new cases. Oral Surg. 35, 54-66. Duhig, J.T. and Ayer, J.P. (1959) Vascular leiomyoma: a study of sixty-one cases. Arch. Pathol. 68, 424-430. Enzinger, F.M., Lattes, R. and Torloni, H. (1969) Histologic Typing of Soft Tissue Tumours. World Health Organization, Geneva, pp. 30-31. Farman, A.G. (1975) Benign smooth muscle tumors. S. Afr. Med. J. 49, 1333-1340 Farman, A.G. and Kay, S. (1977) Oral leiomyosarcoma: report of a case and review of the literature pertaining to smooth muscle tumors of the oral cavity. Oral Surg. 43, 402-409. 9 Erenkel, G. (1965) Auftreten eines Leiomyofibroangioms im Wangen- und Jochbogenbereich. Dtsch Zahnaerztl Z. 20, 168-172. 10 Garrett, J.R. (1969) Angiomyoma of the palate: report of a case. Oral Surg. 27, 103-105. 11 Glas, E. (1905) Beitrage zur Pathologie der Zungengrundtumoren. Wien. Klin. Wochenschr. 18, 746-752. 12 Haedicke, G. and Kaban, L. (1988) Smooth-muscle tumors of the oral cavity. Plast. Reconstr. Surg. 81, 264-269 13 Leung, K.W., Wong, D.Y.K. and Li, W.Y. (1990) Oral leiomyoma: case report. J. Oral Maxillofac. Surg. 48, 735-738. 14 MacDonald, D.G. (1969) Smooth muscle tumors of the mouth. Br. J. Oral Surg. 6, 204-214 15 Merrill, R.G. and Downs, J.R. (1967) Oral leiomyomas: report of two cases. Oral Surg. 23,438-442.
H.S. Kotler et al. /Int. J. Pediatr. Otorhinolaryngol. 29 (1994) 16
17 18 19 20 21 22 23 24 25 26
139-145
145
Miles, A.E.W. and Waterhouse, J.P. (1962) A leiomyosarcoma of the oral cavity with metastasis to lymph-glands. J. Pathol. Bact. 83, 551-555. Natiella, J.R., Neiders, M.E. and Greene, G.W. (1982) Oral leiomyoma: report of six cases and a review of the literature. J. Oral Pathol. II, 353-365. Praal, F.R., Ioannides, CA., van Beek, G.J. and van der Molengraft, F. (1982) Oral leiomyomas. J. Maxillofac. Surg. 10, 229-235. Robbins, S.L., Cotran, R.S. and Kumar, V. (1984) Pathologic Basis of Disease, 3rd Edn. W.B. Saunders Co., Philadelphia, p. 271. Sehrt, E. (1907) Subcutane Leiomyome der Wange und ihre Histologenese. Bruns’ Beitr. Klin. Chir. 54, 723-734 Stout, A.P. (1938) Leiomyoma of the oral cavity. Am. J. Cancer 34, 31-36. Stout, A.P. and Hill, W.T. (1958) Leiomyosarcoma of the superficial soft tissues. Cancer I I, 844-854. Svane, T.J., Smith, B.R., Cosentino, B.J. et al. (1986) Oral leiomyomas: review of the literature and report of a case of palatal angioleiomyoma. J. Peridontol. 57, 433-430. Utz, W. (1965) Beitriige zur Klinik und Pathologic des Leiomyomas in der Mundhiihle. Stoma 18, 190-192. Watabe, T. and Morishima, T. (1963) Rare case of leiomyoma at the root of the tongue in an infant. Otolaryngology (Tokyo) 35, 147-148. Yannopoulos, K. and Stout, A.P. (1962) Smooth muscle tumors in children. Cancer 15, 958-971.