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Intramuscular hemangioma of the mentalis muscle
Intramuscular hemangioma of the mentalis muscle Gregory K. Ingalls, D.D.S., * Gerald J. Bonnington, D.D.S., ** and Allen L. Sisk, D.D.S.,*** Augusta, Ga. MEDICAL
COLLEGE
OF GEORGIA
Hemangiomas of skeletal muscle are relatively uncommon and often difficult to diagnose. In the head and neck, the masseter and trapezius muscles are most commonly reported as being involved. The cause of these lesions is unknown, although theories of development include trauma and abnormal sequestration of embryonic tissue. Treatment has included corticosteroids, sclerosing agents, radiation therapy, embolization. and surgical excision. A review of the literature and the first case report of hemangioma involving the mentalis muscle are presented. (ORAL SURC. ORAL MED. ORAL PATHOL. 60:476-481, 1985)
H
emangiomas involving the skeletal muscles are relatively uncommon and clinically difficult to diagnose. Since the first report of an intramuscular hemangioma by Liston’ in 1843, there have been several reports of hemangiomas involving muscles of the head and neck. Watson and McCarthy2 reviewed 1,308 hemangiomas and reported that 10 (0.8%) were found to be confined to skeletal muscle. Of these, a majority involved the trunk and extremities, presumably because of the large percentage of total body muscle volume in these areas. Jenkins and Delaney,’ in a review of 256 angiomatous tumors of skeletal muscle, found 28 hemangiomas of the head and neck. More recently, Scott4 reviewed 393 cases of intramuscular hemangiomas. Twenty-eight of the lesions occurred in the head and fifteen in the neck, an incidence of 1%. Of the muscles in the head and neck region, hemangioma formation is most commonly reported in the masseter muscle, followed by the trapezius muscle. There have been no previous reports of hemangioma involving the mentalis muscle. The cause of intramuscular hemangioma is unknown. Several theories have been proposed, but the most likely explanation is that the intramuscular hemangioma is a congenital mass, arising by abnormal embryonic sequestrations, similar to congenital *Resident. **Chief Resident. ***Assistant Professor, Surgery.
476
Department
of Oral
and
Maxillofacial
arteriovenous malformations.3,6 The majority of reported casesoccur in young patients. Jenkins and Delaney3reported that 47% of intramuscular hemangiomas occur in the first decadeof life and 94% occur before the end of the third decade. This finding suggests that trauma may play a role. Muscle contractions following trauma, with resultant vascular injury and granulation tissue formation, might contribute to hemangioma development. Hemangiomas were initially labeled as cavernous or capillary; they may now be further described by vessel size. Allen and Enzinger,’ in a review of 89 cases of intramuscular hemangioma, classified the lesions into small vessel, large vessel,or mixed types. The small vessel type was found most commonly in the trunk and upper torso (trunk 32%, head and neck region 30%, upper limb 23%). This type had the shortest clinical history, usually less than 1 year in duration. The mixed type was common in the trunk (53%) and the lower limb (3 l%), was rarely found in the head and neck, and showed the highest recurrence rate (28%). The large vessel type most commonly involved the lower limbs (42%). The incidence of large vessel hemangiomas of the head and neck was 19%, with a recurrence rate of 9%. Clinically, the diagnosis of intramuscular hemangioma is made difficult by the higher incidence of congenital cysts in the younger age groups and malignant lesions among adults.2~4~8 The skin or mucosa overlying the hemangioma will often show increased vascularity, giving the hemangioma a bluish tint. Pulsations, bruits, or thrills are infre-
Volume Number
Intramuscular
60 5
Fig.
1.
hemangioma of mentalis muscle
477
Clinical appearance of the lesion extraorally.
quently associatedwith the intramuscular hemangioma. This is attributed to muscle fiber thickening and fibrosis surrounding and incorporating the lesion. Pain will often be present and is thought to be secondary to compression of adjacent structures by the enlarging mass. Standard radiographs may show evidence of phleboliths, and angiography may be used to reveal the aberrant vessels feeding the lesion. CASEREPORT
On March 3, 1984, a 17-year-old black male patient was referred to one of us (A. S.) for evaluation and treatment of a mass in the chin. The patient stated that he had had a soft, fluctuant mass in his chin for as long as he could remember. The masswas reported to vary in size; increasing following trauma (which the patient sustained frequently while playing football), and then gradually decreasing. On clinical examination, there was a marked soft-tissue swelling in the chin, extending from the midline to the left premolar area. The tissue was soft and rubbery in consistency; no definite outline was recognized. The lesion appeared to be confined to the chin. The inferior border of
the mandible could be palpated through the lesion; it did not appear clinically to be involved. There was no tenderness to palpation; nor was there evidence of fistula or drainage (Fig. 1). There were multiple teeth missing; the mandibular left lateral incisor and both central incisors had been removed approximately 2 years previously. When the lower lip was reflected and stretched taut, a bluish, elevated area could be seen. There were no pulsations, bruits, or thrills. The area was without any tenderness or dysesthesia. No abnormalities were noted on radiographic examination of hard tissue. The only significant finding in the patient’s past medical history was the presence of a cystic hygroma at birth. When the patient was 4 years old the cystic hygroma was removed in four surgical procedures. Laboratory values (SMA-6, CBC, and urinalysis) were all within normal limits. On the basis of the history of long duration of the lesion and recurrent swelling secondary to trauma, the clinical findings of bluish discoloration of overlying oral mucosa, the lack of detectable pulsations, thrills, or bruits, and the lack of positive radiographic findings, a provisional diagnosis of hemangioma was made. Diagnoses of lymphangioma and arteriovenous malformation were also considered. Because of the provisional diagnosis of a vascular lesion, arteriography was considered to aid in diagnosis and to
478 Ingalls, Bonnington, and Sisk
Oral Surg. November, 1985
Fig. 2. Arteriogram of left common carotid artery showing aberrant branch of the facial artery extending into the lesion. (Subtraction film, lateral view.) identify the extent of the lesion and contributions to the lesion from surrounding blood vessels. On March 6, 1984, the patient underwent bilateral common carotid arteriography via the right femoral artery. Selective injections were made into the right external carotid artery and into both common carotid arteries just below their bifurcations. The left common carotid injection revealed a collection of tortuous vesselsof varying caliber just beneath the left side of the anterior mandible, extending to the midline. An aberrant branch of the left facial artery extended from the facial artery into the area of the lesion (Figs. 2 and 3). The right carotid artery injections revealed no abnormalities. The tentative clinical diagnosis of hemangioma was made. On the following day the patient was taken to the operating room where, after induction of general anesthesia, the hemangioma was resected without complication. The aberrant branch of the facial artery was identified and ligated from an intraoral approach. The lesion was noted to involve the mentalis muscle and the left mental nerve. The
postoperative course was uneventful, and the patient was discharged on the sixth hospital day. Histologically, the lesion was described as consisting of numerous fasicles of striated skeletal muscle fibers supported by a fibro fatty stroma. Throughout the specimen and interspersed between the muscle fibers were numerous cavernous endothelium-lined spaces that were engorged with erythrocytes and lined with a well-defined smooth muscle wall (Figs. 4 and 5). The diagnosis of intramuscular hemangioma was made. DISCUSSION
Trauma has been implicated in the pathogen&s of intramuscular hemangiomas. In this case,the patient was a healthy, active male who had been participating in contact sports most of his life. He reported having received numerous blows to the chin area over the years. Treatment of intramuscular hemangiomas has
Volume Number
Intramuscular hemangioma of mentalis muscle 479
60 5
Fig.
3. Arteriogram. (Subtraction film, posteroanterior view.)
been extensively reported.*,3,8-19Treatment has included the use of systemic corticosteroids, injection of sclerosing agents, radiation therapy, and surgical excision. The extensive anastomosis of contributing blood vesselsto the intramuscular hemangioma may be responsible for the rare complication of intravascular coagulation at the time of surgical excision.18 The currently accepted treatment to minimize the complications, as well as to halt the destruction of surrounding tissues, is total excision of the lesion with a margin of normal muscle. It should be noted, however, that there may be minor arterial feeder vessels that are not visible on preoperative angiograms and that may cause excessive blood loss during surgery. These minor feeder vesselsmay also
be responsible for recurrence of the hemangiomas.8.I3 Another treatment that has considerable reported success is the use of embolization. In theory, a spontaneous thrombosis of the vessel distal to the embolus effectively seals off the aberrant feeder vessel. Bennett and Zook19have reported the injection of fragments of sternocleidomastoid muscle into the external carotid system, and Schrudde and PetroL vici” more recently described the use of small particles of Gelfoam or lyophilized dura mater. Embolization was not considered in our casebecause the caliber of the feeding vessel was reIatively small and the vessel was positioned to allow an intraoral surgical approach for ligation. Our surgical
480
Ingalls, Bennington, and Sisk
Oral Surg. November. I985
Fig. 4. The lesion consists of numerous fascicles of striated muscle, with numerous cavernous endotheliurn-lined spaces.(Hematoxylin and eosin stain. Magnification, X100.)
Fig. 5. Cavernous endothelium-lined spaces engorged with erythrocytes and lined with well-defined smooth muscle walls. (Hematoxylin and eosin stain. Magnification, X 100.)
approach was via an incision made at the mucogingival junction, with submucosal and supraperiosteal dissection. This allowed adequate exposure for access and ligation of the aberrant branch of the facial artery and removal of the hemangioma without a cosmetic deficit. REFERENCES 1. Liston R: Case of erectile tumor in the popliteal space. Med Chir Trans 26: 120-132, 1843. 2. Watson WL, McCarthy WD: Blood and lymph vessel tumors: a report of 1,056 cases. Surg Gynecol Obstet 71: 569-588, 1940. 3. Jenkins HP, Delaney PA: Benign angiomatous tumors of skeletal muscles. Surg Gynecol Obstet 55: 464-480, 1932.
4. Scott JES: Hemanaiomas in skeletal muscle. Br J Surg 44: 496-501, 1957. 5. Clemis JD, Briggs DR, Changus GW: Intramuscular hemangioma in the head and neck. Can J Otolaryngol 4: 339-347, 1975. 6. Welsh D, Hengerer AS: The diagnosis and treatment of intramuscular hemangiomas of the masseter muscle. Am J Otolaryngol I: 186-191. 1980. 7. Allen PW, Enzinger FM: Hemangioma of skeletal muscle: an analysis of 89 cases. Cancer 29: S-22, 1972. 8. Willis RA: Pathology of Tumours, ed. 4, London, 1967, Butterworth & Company, Ltd., pp 700-717. 9. Makino Y: A clinicopathological study on soft tissue tumors of the head and neck. Acta Path01 Jpn 29: 389-408, 1979. 10. Goldwyn RM: Soft tissue swellings. Practitioner 212: 185192, 1914. 11. Gruss JS: Hemangioma of the mandible: a case report. Br J Oral Surg 12: 24-32, 1974.
Volume 60 Number 5 12. Lund BA, Dahhn DC: Hemangiomas of the mandible and maxilla. J Oral Surg 22: 234-242, 1964. 13. Furgusson ILC: Hemangioma of skeletal muscle. Br J Surg 59: 634-631, 1972. 14. Conley JJ, Clairmont AA: Intramuscular hemangioma of the masseter muscle: case report. Plast Reconstr Surg 60: 121124, 1977. 15. Merchant NE Cavernous hemangioma of the cheek. Dent Pratt 22: 436, 1912. 16. Briggs DR, Porubsky ES, Teeslink R, Sharbaugh AH: Intramuscular hemangioma of the neck. Tram Am Acad Ophth Otolaryngol 84: 148-151, 1977. 17. Schrudde J, Petrovici V: Surgical treatment of giant hemangioma of the facial region after arterial embolization. Plast Reconstr Surg 68: 878-889, 1981.
intramuscular
hemangioma of mentalis muscle
481
18. Ibrahim AA, Dale WA: Disseminated intravascular coagulation after surgical excision of cavernous hemangioma. South Med J 7t: 760-761, 1979. 19. Bennett JE, Zook EG: Treatment of arteriovenous fistulas in cavernous hemangiomas of face by muscle embolization: case report. Plast Reconstr Surg 50: 84-87, 1972. Reprint requests to: Dr. Allen L. Sisk AD-I 18 Department of Oral and Maxillofacial Medical College of Georgia Augusta. GA 309 I2
Surgery
COLLEGE
OF GEORGIA
Hemangiomas of skeletal muscle are relatively uncommon and often difficult to diagnose. In the head and neck, the masseter and trapezius muscles are most commonly reported as being involved. The cause of these lesions is unknown, although theories of development include trauma and abnormal sequestration of embryonic tissue. Treatment has included corticosteroids, sclerosing agents, radiation therapy, embolization. and surgical excision. A review of the literature and the first case report of hemangioma involving the mentalis muscle are presented. (ORAL SURC. ORAL MED. ORAL PATHOL. 60:476-481, 1985)
H
emangiomas involving the skeletal muscles are relatively uncommon and clinically difficult to diagnose. Since the first report of an intramuscular hemangioma by Liston’ in 1843, there have been several reports of hemangiomas involving muscles of the head and neck. Watson and McCarthy2 reviewed 1,308 hemangiomas and reported that 10 (0.8%) were found to be confined to skeletal muscle. Of these, a majority involved the trunk and extremities, presumably because of the large percentage of total body muscle volume in these areas. Jenkins and Delaney,’ in a review of 256 angiomatous tumors of skeletal muscle, found 28 hemangiomas of the head and neck. More recently, Scott4 reviewed 393 cases of intramuscular hemangiomas. Twenty-eight of the lesions occurred in the head and fifteen in the neck, an incidence of 1%. Of the muscles in the head and neck region, hemangioma formation is most commonly reported in the masseter muscle, followed by the trapezius muscle. There have been no previous reports of hemangioma involving the mentalis muscle. The cause of intramuscular hemangioma is unknown. Several theories have been proposed, but the most likely explanation is that the intramuscular hemangioma is a congenital mass, arising by abnormal embryonic sequestrations, similar to congenital *Resident. **Chief Resident. ***Assistant Professor, Surgery.
476
Department
of Oral
and
Maxillofacial
arteriovenous malformations.3,6 The majority of reported casesoccur in young patients. Jenkins and Delaney3reported that 47% of intramuscular hemangiomas occur in the first decadeof life and 94% occur before the end of the third decade. This finding suggests that trauma may play a role. Muscle contractions following trauma, with resultant vascular injury and granulation tissue formation, might contribute to hemangioma development. Hemangiomas were initially labeled as cavernous or capillary; they may now be further described by vessel size. Allen and Enzinger,’ in a review of 89 cases of intramuscular hemangioma, classified the lesions into small vessel, large vessel,or mixed types. The small vessel type was found most commonly in the trunk and upper torso (trunk 32%, head and neck region 30%, upper limb 23%). This type had the shortest clinical history, usually less than 1 year in duration. The mixed type was common in the trunk (53%) and the lower limb (3 l%), was rarely found in the head and neck, and showed the highest recurrence rate (28%). The large vessel type most commonly involved the lower limbs (42%). The incidence of large vessel hemangiomas of the head and neck was 19%, with a recurrence rate of 9%. Clinically, the diagnosis of intramuscular hemangioma is made difficult by the higher incidence of congenital cysts in the younger age groups and malignant lesions among adults.2~4~8 The skin or mucosa overlying the hemangioma will often show increased vascularity, giving the hemangioma a bluish tint. Pulsations, bruits, or thrills are infre-
Volume Number
Intramuscular
60 5
Fig.
1.
hemangioma of mentalis muscle
477
Clinical appearance of the lesion extraorally.
quently associatedwith the intramuscular hemangioma. This is attributed to muscle fiber thickening and fibrosis surrounding and incorporating the lesion. Pain will often be present and is thought to be secondary to compression of adjacent structures by the enlarging mass. Standard radiographs may show evidence of phleboliths, and angiography may be used to reveal the aberrant vessels feeding the lesion. CASEREPORT
On March 3, 1984, a 17-year-old black male patient was referred to one of us (A. S.) for evaluation and treatment of a mass in the chin. The patient stated that he had had a soft, fluctuant mass in his chin for as long as he could remember. The masswas reported to vary in size; increasing following trauma (which the patient sustained frequently while playing football), and then gradually decreasing. On clinical examination, there was a marked soft-tissue swelling in the chin, extending from the midline to the left premolar area. The tissue was soft and rubbery in consistency; no definite outline was recognized. The lesion appeared to be confined to the chin. The inferior border of
the mandible could be palpated through the lesion; it did not appear clinically to be involved. There was no tenderness to palpation; nor was there evidence of fistula or drainage (Fig. 1). There were multiple teeth missing; the mandibular left lateral incisor and both central incisors had been removed approximately 2 years previously. When the lower lip was reflected and stretched taut, a bluish, elevated area could be seen. There were no pulsations, bruits, or thrills. The area was without any tenderness or dysesthesia. No abnormalities were noted on radiographic examination of hard tissue. The only significant finding in the patient’s past medical history was the presence of a cystic hygroma at birth. When the patient was 4 years old the cystic hygroma was removed in four surgical procedures. Laboratory values (SMA-6, CBC, and urinalysis) were all within normal limits. On the basis of the history of long duration of the lesion and recurrent swelling secondary to trauma, the clinical findings of bluish discoloration of overlying oral mucosa, the lack of detectable pulsations, thrills, or bruits, and the lack of positive radiographic findings, a provisional diagnosis of hemangioma was made. Diagnoses of lymphangioma and arteriovenous malformation were also considered. Because of the provisional diagnosis of a vascular lesion, arteriography was considered to aid in diagnosis and to
478 Ingalls, Bonnington, and Sisk
Oral Surg. November, 1985
Fig. 2. Arteriogram of left common carotid artery showing aberrant branch of the facial artery extending into the lesion. (Subtraction film, lateral view.) identify the extent of the lesion and contributions to the lesion from surrounding blood vessels. On March 6, 1984, the patient underwent bilateral common carotid arteriography via the right femoral artery. Selective injections were made into the right external carotid artery and into both common carotid arteries just below their bifurcations. The left common carotid injection revealed a collection of tortuous vesselsof varying caliber just beneath the left side of the anterior mandible, extending to the midline. An aberrant branch of the left facial artery extended from the facial artery into the area of the lesion (Figs. 2 and 3). The right carotid artery injections revealed no abnormalities. The tentative clinical diagnosis of hemangioma was made. On the following day the patient was taken to the operating room where, after induction of general anesthesia, the hemangioma was resected without complication. The aberrant branch of the facial artery was identified and ligated from an intraoral approach. The lesion was noted to involve the mentalis muscle and the left mental nerve. The
postoperative course was uneventful, and the patient was discharged on the sixth hospital day. Histologically, the lesion was described as consisting of numerous fasicles of striated skeletal muscle fibers supported by a fibro fatty stroma. Throughout the specimen and interspersed between the muscle fibers were numerous cavernous endothelium-lined spaces that were engorged with erythrocytes and lined with a well-defined smooth muscle wall (Figs. 4 and 5). The diagnosis of intramuscular hemangioma was made. DISCUSSION
Trauma has been implicated in the pathogen&s of intramuscular hemangiomas. In this case,the patient was a healthy, active male who had been participating in contact sports most of his life. He reported having received numerous blows to the chin area over the years. Treatment of intramuscular hemangiomas has
Volume Number
Intramuscular hemangioma of mentalis muscle 479
60 5
Fig.
3. Arteriogram. (Subtraction film, posteroanterior view.)
been extensively reported.*,3,8-19Treatment has included the use of systemic corticosteroids, injection of sclerosing agents, radiation therapy, and surgical excision. The extensive anastomosis of contributing blood vesselsto the intramuscular hemangioma may be responsible for the rare complication of intravascular coagulation at the time of surgical excision.18 The currently accepted treatment to minimize the complications, as well as to halt the destruction of surrounding tissues, is total excision of the lesion with a margin of normal muscle. It should be noted, however, that there may be minor arterial feeder vessels that are not visible on preoperative angiograms and that may cause excessive blood loss during surgery. These minor feeder vesselsmay also
be responsible for recurrence of the hemangiomas.8.I3 Another treatment that has considerable reported success is the use of embolization. In theory, a spontaneous thrombosis of the vessel distal to the embolus effectively seals off the aberrant feeder vessel. Bennett and Zook19have reported the injection of fragments of sternocleidomastoid muscle into the external carotid system, and Schrudde and PetroL vici” more recently described the use of small particles of Gelfoam or lyophilized dura mater. Embolization was not considered in our casebecause the caliber of the feeding vessel was reIatively small and the vessel was positioned to allow an intraoral surgical approach for ligation. Our surgical
480
Ingalls, Bennington, and Sisk
Oral Surg. November. I985
Fig. 4. The lesion consists of numerous fascicles of striated muscle, with numerous cavernous endotheliurn-lined spaces.(Hematoxylin and eosin stain. Magnification, X100.)
Fig. 5. Cavernous endothelium-lined spaces engorged with erythrocytes and lined with well-defined smooth muscle walls. (Hematoxylin and eosin stain. Magnification, X 100.)
approach was via an incision made at the mucogingival junction, with submucosal and supraperiosteal dissection. This allowed adequate exposure for access and ligation of the aberrant branch of the facial artery and removal of the hemangioma without a cosmetic deficit. REFERENCES 1. Liston R: Case of erectile tumor in the popliteal space. Med Chir Trans 26: 120-132, 1843. 2. Watson WL, McCarthy WD: Blood and lymph vessel tumors: a report of 1,056 cases. Surg Gynecol Obstet 71: 569-588, 1940. 3. Jenkins HP, Delaney PA: Benign angiomatous tumors of skeletal muscles. Surg Gynecol Obstet 55: 464-480, 1932.
4. Scott JES: Hemanaiomas in skeletal muscle. Br J Surg 44: 496-501, 1957. 5. Clemis JD, Briggs DR, Changus GW: Intramuscular hemangioma in the head and neck. Can J Otolaryngol 4: 339-347, 1975. 6. Welsh D, Hengerer AS: The diagnosis and treatment of intramuscular hemangiomas of the masseter muscle. Am J Otolaryngol I: 186-191. 1980. 7. Allen PW, Enzinger FM: Hemangioma of skeletal muscle: an analysis of 89 cases. Cancer 29: S-22, 1972. 8. Willis RA: Pathology of Tumours, ed. 4, London, 1967, Butterworth & Company, Ltd., pp 700-717. 9. Makino Y: A clinicopathological study on soft tissue tumors of the head and neck. Acta Path01 Jpn 29: 389-408, 1979. 10. Goldwyn RM: Soft tissue swellings. Practitioner 212: 185192, 1914. 11. Gruss JS: Hemangioma of the mandible: a case report. Br J Oral Surg 12: 24-32, 1974.
Volume 60 Number 5 12. Lund BA, Dahhn DC: Hemangiomas of the mandible and maxilla. J Oral Surg 22: 234-242, 1964. 13. Furgusson ILC: Hemangioma of skeletal muscle. Br J Surg 59: 634-631, 1972. 14. Conley JJ, Clairmont AA: Intramuscular hemangioma of the masseter muscle: case report. Plast Reconstr Surg 60: 121124, 1977. 15. Merchant NE Cavernous hemangioma of the cheek. Dent Pratt 22: 436, 1912. 16. Briggs DR, Porubsky ES, Teeslink R, Sharbaugh AH: Intramuscular hemangioma of the neck. Tram Am Acad Ophth Otolaryngol 84: 148-151, 1977. 17. Schrudde J, Petrovici V: Surgical treatment of giant hemangioma of the facial region after arterial embolization. Plast Reconstr Surg 68: 878-889, 1981.
intramuscular
hemangioma of mentalis muscle
481
18. Ibrahim AA, Dale WA: Disseminated intravascular coagulation after surgical excision of cavernous hemangioma. South Med J 7t: 760-761, 1979. 19. Bennett JE, Zook EG: Treatment of arteriovenous fistulas in cavernous hemangiomas of face by muscle embolization: case report. Plast Reconstr Surg 50: 84-87, 1972. Reprint requests to: Dr. Allen L. Sisk AD-I 18 Department of Oral and Maxillofacial Medical College of Georgia Augusta. GA 309 I2
Surgery