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Intramuscular hemangioma of masseter muscle with prominent formation of phleboliths: A case report
Auris Nasus Larynx 35 (2008) 587–591 www.elsevier.com/locate/anl
Intramuscular hemangioma of masseter muscle with prominent formation of phleboliths: A case report Hiroaki Kanaya *, Yutaka Saito, Nobuyasu Gama, Wataru Konno, Hideki Hirabayashi, Shin-ichi Haruna Department of Otorhinolaryngology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Tochigi 321-0293, Japan Received 14 August 2007; accepted 3 November 2007 Available online 18 January 2008
Abstract A 14-year-old girl was referred for evaluation and management of progressive, painful swelling of the right cheek. Swelling had been present since 3-year old and had gradually increased in size. Magnetic resonance imaging (MRI) revealed a well-circumscribed, brighter mass in the right masseter muscle with numerous rounded areas of signal hypointensity. Preoperative diagnosis was intramuscular hemangioma of the masseter muscle and surgery was performed. The tumor was completely removed except for a few, small phleboliths. Pathological examination of resected tissue led to a high suspicion of cavernous hemangioma with phlebolithiasis. Nine months postoperatively, the patient developed another painful mass in the right masseter muscle. MRI indicated recurrent hemangioma and further surgery was performed. Careful exploration resulted in completely removal of residual phleboliths accompanied with fibroadipose tissue. Part of the buccal branch of the facial nerve was excised to achieve complete resection of the lesion. Histological examination revealed distinct venous formation in phleboliths adjacent to fibroadipose tissue, demonstrating that both phleboliths and feeding vessels had been left by the previous operation. The present report reviews the literature on intramuscular hemangiomas of the masseter muscle, and discusses diagnostic methods and optimal surgical treatment. # 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: Intramuscular hemangioma; Masseter muscle; Phlebolith formation
1. Introduction Intramuscular hemangioma is a relatively rare, benign tumor of vascular origin, accounting for less than 1% of all hemangiomas [1]. These tumors occur most often in the trunk and extremities, with the head and neck region accounting for only 10–20% of intramuscular hemangiomas, and while any muscle may be affected, the masseter muscle is the most frequently involved site [2]. Half of these tumors are clinically apparent in the first decade of life. The presence of a slowly enlarging and often painful mass is the predominant complaint. No gender predispositions are seen. * Corresponding author. Tel.: +81 282 86 1111x2652; fax: +81 282 86 5928. E-mail address: [email protected] (H. Kanaya).
On physical examination, the overlying skin is generally normal, but may display reddish blue discoloration or even be hyperthermic. Compressibility, pulsation, thrills and bruits anticipated in such vascular lesions are usually absent because of thickening and fibrosis of the muscular fibers surrounded and involved with tumor [1]. Given the rarity of these tumors, deep location and variable clinical presentation, the diagnosis of intramuscular hemangioma could be difficult [3–6]. Furthermore, unnecessary risk to the facial nerve often occurs when tumors are present in the face. Phlebolith formation and calcification have been reported as characteristic features of hemangiomas [7–9], so the current availability of diagnostic imaging modalities such as computed tomography (CT), magnetic resonance imaging (MRI) [10,11] and ultrasonography has significantly increased the accuracy of preoperative diagnosis of patients
0385-8146/$ – see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.anl.2007.11.003
588
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
with this lesion. We herein report a rare case of intramuscular hemangioma of the masseter muscle with prominent formation of phleboliths. In this case, we encountered difficulties in surgical treatment due to insufficient procedure on the first operation.
2. Case report A 14-year-old girl was referred for evaluation and management of progressive, painful swelling of the right cheek. The swelling had been present since 3-year old and had gradually increased in size. No prior history of trauma, dental problems, trismus or other medical problems was elicited. Clinical examination showed a roughly 4-cm 3-cm firm, mobile, mildly compressible mass in the right masseter muscle. No pulsation, thrills or bruits were noted. Several hard, round nodules were palpable under the skin, and skin covering the lesion appeared normal. The patient was otherwise asymptomatic. T2-weighted coronal imaging revealed a well-circumscribed, hyperintense mass in the right masseter muscle with numerous rounded areas of signal hypointensity (Fig. 1A). Preoperative diagnosis was hemangioma of the masseter muscle and surgery was scheduled. Using general anesthesia and endotracheal intubation, an incision about 10 cm long was made in the right retromandibular area. Dissection was performed anteriorly and superiorly just superficial to the masseter muscle. The facial artery and vein were clamped, and the marginal mandibular branch of the facial nerve was clearly identified and preserved. Dissection was continued to the area where most of the swelling was apparent on clinical examination. Careful blunt dissection between masseter muscle fibers revealed dark-
brown hemangioma filled with numerous large and small phleboliths. No large vascular connections to the tumor were noted from the masseter muscle or adjacent fibroadipose tissue, and no vascular formation was observed on the tumor surface. This condition was attributed to most of the vascular lumina of the tumor already being obstructed and obliterated due to prominent phlebolith formation. Although most of the tumor was removed (Fig. 2A), a few small phleboliths were found deep to the masseter muscle. As the buccal branch of the facial nerve was tightly wound around these phleboliths, the decision was made to leave that these structures in place to avoid facial palsy. Wound closure was accomplished without difficulty. Blood loss was <200 ml during operation. On pathological examination of the resected tissue, cavernous hemangioma with marked phlebolithiasis was highly suspected. A total of 21 brown or grayish-yellow phleboliths were found within the resected tumor (Fig. 2B). Microscopically, each phlebolith revealed an apparent concentric structure comprising red blood cells and calcified fibrinous material (Fig. 3A). Equivocal vascular lumens were observed between phleboliths and muscular fibers, and endothelial cells were completely flattened by compression (Fig. 3B). Nine months postoperatively, the patient developed another painful mass in the right masseter muscle. MRI revealed recurrent hemangioma (Fig. 1B) and reoperation was performed. Under general anesthesia, the incision was made overlapping the scar of the first operation and extended to the tragus of the ear by means of parotidectomy. Postoperative scar formation was gently dissected and the masseter muscle exposed. Following careful exploration, residual phleboliths were completely removed accompanied by fibroadipose tissue (Fig. 4A). Fine vascular connections were observed and completely clamped. Part of the buccal
Fig. 1. (A) Coronal view on preoperative MRI (T2-weighted) showing signal hyperintensity in the right masseter muscle with many round, hypointense areas (arrow). (B) Coronal view on postoperative T2-weighted MRI revealing recurrent hemangioma in right masseter muscle (arrow).
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
Fig. 2. (A) Surgically resected tissue was filled with numerous, round to oval-shaped phleboliths (bar, 1 cm). (B) A total of 21 dark brown phleboliths up to 12 mm in diameter, were observed within the tumor (bar, 1 cm).
branch of the facial nerve was excised to accomplish complete resection of the lesion. To prevent recurrence, normal muscular tissue was also removed well beyond the gross limit (Fig. 4B). Histological examination revealed
589
Fig. 4. (A) Resected phleboliths were accompanied by fibroadipose tissue (bar, 1 cm). (B) After complete removal of recurrent tumor (*parotid gland). Substantial right masseter muscle was resected (arrows).
typical cavernous hemangioma in fibroadipose tissue adjacent to phleboliths, demonstrating that both phleboliths and feeding vessels remained from the previous operation. As of 1 year after reoperation, facial asymmetry had spontaneously improved and no evidence of recurrent disease was seen.
Fig. 3. (A) Histopathology showing prominent phlebolith formation with apparent concentric structure. Lamellated red blood cells and fibrinous materials were seen in phleboliths (10, hematoxylin and eosin (HE)). (B) Equivocal vascular lumens (arrow) were observed between phleboliths (*) and muscular fibers (40, HE).
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H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
3. Discussion In general, diagnosis of intramuscular hemangioma of the head and neck region can be difficult [4–6]. The lack of objective clinical findings (e.g., compressibility, pulsation, thrills, bruits, evident skin discoloration) and the rare incidence of this lesion allows ample opportunity for misdiagnosis. As only 8% of all cases of intramuscular hemangiomas are diagnosed pre-operatively [5], a variety of parotid and muscular neoplasms, benign muscular hypertrophy, congenital cysts and lymphadenopathies are commonly confused in the differential diagnosis [6]. Phlebolith formation and calcification have been reported as a characteristic feature of hemangiomas [7–9], so diagnostic imaging modalities like CT, MRI [10,11] and ultrasonography have significantly increased the accuracy of preoperative diagnosis for this lesion. Intramuscular hemangiomas are characteristically much brighter on T2- than T1-weighted imaging due to the increased free water present within stagnant blood in the vessels. In this case, prominent phlebolith formation on T2-weighted imaging led to appropriate preoperative diagnosis. Phlebolith formation within intramuscular hemangioma is present in approximately 25% of cases [7,8]. The pathogenesis of phleboliths [12,13] is thought to involve thrombi produced by slowing of peripheral blood flow becoming organized and mineralized. The concentric structure on the cut surface of the phlebolith with histopathology suggests the following growth mechanism. Initially, calcification of the thrombus occurs, forming the core of the phlebolith. The fibrinous component then undergoes secondary calcification and becomes attached. Repetition of this process causes enlargement of the phlebolith. In our patient, long-term presence of hemangioma and stagnant blood flow caused a large number of phleboliths. Many forms of therapy have been advocated to cure or control intramuscular hemangioma [3,11]. Sclerosing agents provide a palliative treatment for intramuscular hemangioma, but post-injection scarring and recurrence have limited their administration. Systemic steroids have been used successfully in treatment of cutaneous capillary hemangiomas [11]. In our case, however, administration of sclerosing agents and/or systemic steroids would not be effective because blood flow in the tumor vessels has already stagnated by prominent phlebolith formation. The ideal treatment is complete excision of the tumor with a surrounding margin of normal muscular tissue, due to the infiltrative nature of the tumor [6,14–16]. Minor feeding vessels and residual tumor are responsible for a recurrence rate of approximately 18%, with 7% recurring more than once [2]. In our experience with this case, a few phleboliths were left in the first operation due to the unnecessary risks of facial palsy. Incomplete removal was followed by recurrences and deeper invasion of adjacent
tissues by tumor. As a result of recurrence, we had to perform reoperation with extended skin incision and deeper dissection in scarred muscular tissue. Phleboliths were certainly observed within the tumor tissue histologically, so careful exploration and complete extirpation of phleboliths is to be strongly recommended even if vascular formations or connecting vessels are not evident near the phleboliths. Excessive blood loss during surgery may result in significant surgical morbidity. Preoperative arterial embolization can be invaluable in diminishing blood loss and is thus widely used during surgery on vascular tumors. However, Ichimura et al. [14] noted that complete tumor resection could be performed with less bleeding if a wide surgical field exposing the whole tumor and surrounding area was obtained. Ligation of feeding vessels also helps to minimize blood loss. In practice, ligating the masseteric branch of the facial artery is sufficient, as most intramuscular hemangiomas including the present case arise in the lower part of the masseter muscle [14,17]. Prominent formation of phleboliths had already decreased blood flow, and ligation of the facial artery in the first operation diminished extensive blood loss in our surgery.
References [1] Barnes L. Tumors and tumor like lesions of the soft tissue. In: Barnes L, editor. Pathology of the head and neck. 2nd ed., New York, NY: Marcel Dekker; 2001. p. 900–1. [2] Allen PW, Enzinger FM. Hemangioma of skeletal muscle. An analysis of 89 cases. Cancer 1972;29:8–22. [3] Wolf GT, Daniel F, Krause CJ, Kaufman RS. Intramuscular hemangioma of the head and neck. Laryngoscope 1985;95:210–3. [4] Elahi MM, Parnes L, Fox A. Hemangioma of the masseter muscle. J Otolaryngol 1992;21:177–9. [5] Avci G, Yim S, Misirliogolu A, Akoz T, Kartal LK. Intramasseteric hemangioma. Plast Reconst Surg 2002;109:1748–9. [6] Demir Z, Oktem F, Celebioglu S. Rare case of intramasseteric cavernous hemangioma in a three-year-old boy: a diagnostic dilemma. Ann Otol Rhinol Laryngol 2004;113:455–8. [7] Morris SJ, Adams H. Case report: paediatric intramuscular haemangiomata—don’t overlook the phlebolith! Br J Radiol 1995;68: 208–11. [8] Osada K, Yoshihara T, Itoh M. Intramasseter hemangiomas: a case report. J Otolaryngol 2000;29:166–9. [9] Mandel L. Intramuscular hemangioma with phleboliths or choristoma? J Oral Maxillofac Surg 2001;59:1511–2. [10] Yonetsu K, Nakayama E, Miwa K, Tanaka T, Araki K, Kanda S, et al. Magnetic resonance imaging of oral and maxillofacial angiomas. Oral Surg Oral Med Oral Pathol 1993;76:783–9. [11] Rossiter JL, Hendrix RA, Tom LWC, Potsic WP. Intramuscular hemangioma of the head and neck. Otolaryngol Head Neck Surg 1993;108:18–26. [12] Sano K, Ogawa A, Inokuchi T, Takahashi H, Hisatsune K, Eng D. Buccal hemangioma with phleboliths; report of two cases. Oral Surg Oral Med Oral Pathol 1988;65:151–6. [13] Raymond AK, Batsakis JG. Pathology consultation: angiolithiasis and sialolithiasis in the head and neck. Ann Otol Rhinol Laryngol 1992;101:455–7.
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591 [14] Ichimura K, Nibu K, Tanaka T. Essentials of surgical treatment for intramasseteric hemangioma. Eur Arch Otorhinolaryngol 1995;252: 125–9. [15] Welsh D, Hengerer AS. The diagnosis and treatment of intramuscular hemangiomas of the masseter muscle. Am J Otolaryngol 1980;1:186–90.
591
[16] Hart B, Schwartz HC. Cavernous hemangioma of the masseter muscle: report of a case. J Oral Maxillofac Surg 1995;53: 467–9. [17] Ichimura K, Tanaka T, Kitahara N. Surgery of the masseter muscle. Pract Otol (Kyoto) 1989;82:1209–19.
Intramuscular hemangioma of masseter muscle with prominent formation of phleboliths: A case report Hiroaki Kanaya *, Yutaka Saito, Nobuyasu Gama, Wataru Konno, Hideki Hirabayashi, Shin-ichi Haruna Department of Otorhinolaryngology, Dokkyo Medical University, 880 Kitakobayashi, Mibu, Tochigi 321-0293, Japan Received 14 August 2007; accepted 3 November 2007 Available online 18 January 2008
Abstract A 14-year-old girl was referred for evaluation and management of progressive, painful swelling of the right cheek. Swelling had been present since 3-year old and had gradually increased in size. Magnetic resonance imaging (MRI) revealed a well-circumscribed, brighter mass in the right masseter muscle with numerous rounded areas of signal hypointensity. Preoperative diagnosis was intramuscular hemangioma of the masseter muscle and surgery was performed. The tumor was completely removed except for a few, small phleboliths. Pathological examination of resected tissue led to a high suspicion of cavernous hemangioma with phlebolithiasis. Nine months postoperatively, the patient developed another painful mass in the right masseter muscle. MRI indicated recurrent hemangioma and further surgery was performed. Careful exploration resulted in completely removal of residual phleboliths accompanied with fibroadipose tissue. Part of the buccal branch of the facial nerve was excised to achieve complete resection of the lesion. Histological examination revealed distinct venous formation in phleboliths adjacent to fibroadipose tissue, demonstrating that both phleboliths and feeding vessels had been left by the previous operation. The present report reviews the literature on intramuscular hemangiomas of the masseter muscle, and discusses diagnostic methods and optimal surgical treatment. # 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: Intramuscular hemangioma; Masseter muscle; Phlebolith formation
1. Introduction Intramuscular hemangioma is a relatively rare, benign tumor of vascular origin, accounting for less than 1% of all hemangiomas [1]. These tumors occur most often in the trunk and extremities, with the head and neck region accounting for only 10–20% of intramuscular hemangiomas, and while any muscle may be affected, the masseter muscle is the most frequently involved site [2]. Half of these tumors are clinically apparent in the first decade of life. The presence of a slowly enlarging and often painful mass is the predominant complaint. No gender predispositions are seen. * Corresponding author. Tel.: +81 282 86 1111x2652; fax: +81 282 86 5928. E-mail address: [email protected] (H. Kanaya).
On physical examination, the overlying skin is generally normal, but may display reddish blue discoloration or even be hyperthermic. Compressibility, pulsation, thrills and bruits anticipated in such vascular lesions are usually absent because of thickening and fibrosis of the muscular fibers surrounded and involved with tumor [1]. Given the rarity of these tumors, deep location and variable clinical presentation, the diagnosis of intramuscular hemangioma could be difficult [3–6]. Furthermore, unnecessary risk to the facial nerve often occurs when tumors are present in the face. Phlebolith formation and calcification have been reported as characteristic features of hemangiomas [7–9], so the current availability of diagnostic imaging modalities such as computed tomography (CT), magnetic resonance imaging (MRI) [10,11] and ultrasonography has significantly increased the accuracy of preoperative diagnosis of patients
0385-8146/$ – see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.anl.2007.11.003
588
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
with this lesion. We herein report a rare case of intramuscular hemangioma of the masseter muscle with prominent formation of phleboliths. In this case, we encountered difficulties in surgical treatment due to insufficient procedure on the first operation.
2. Case report A 14-year-old girl was referred for evaluation and management of progressive, painful swelling of the right cheek. The swelling had been present since 3-year old and had gradually increased in size. No prior history of trauma, dental problems, trismus or other medical problems was elicited. Clinical examination showed a roughly 4-cm 3-cm firm, mobile, mildly compressible mass in the right masseter muscle. No pulsation, thrills or bruits were noted. Several hard, round nodules were palpable under the skin, and skin covering the lesion appeared normal. The patient was otherwise asymptomatic. T2-weighted coronal imaging revealed a well-circumscribed, hyperintense mass in the right masseter muscle with numerous rounded areas of signal hypointensity (Fig. 1A). Preoperative diagnosis was hemangioma of the masseter muscle and surgery was scheduled. Using general anesthesia and endotracheal intubation, an incision about 10 cm long was made in the right retromandibular area. Dissection was performed anteriorly and superiorly just superficial to the masseter muscle. The facial artery and vein were clamped, and the marginal mandibular branch of the facial nerve was clearly identified and preserved. Dissection was continued to the area where most of the swelling was apparent on clinical examination. Careful blunt dissection between masseter muscle fibers revealed dark-
brown hemangioma filled with numerous large and small phleboliths. No large vascular connections to the tumor were noted from the masseter muscle or adjacent fibroadipose tissue, and no vascular formation was observed on the tumor surface. This condition was attributed to most of the vascular lumina of the tumor already being obstructed and obliterated due to prominent phlebolith formation. Although most of the tumor was removed (Fig. 2A), a few small phleboliths were found deep to the masseter muscle. As the buccal branch of the facial nerve was tightly wound around these phleboliths, the decision was made to leave that these structures in place to avoid facial palsy. Wound closure was accomplished without difficulty. Blood loss was <200 ml during operation. On pathological examination of the resected tissue, cavernous hemangioma with marked phlebolithiasis was highly suspected. A total of 21 brown or grayish-yellow phleboliths were found within the resected tumor (Fig. 2B). Microscopically, each phlebolith revealed an apparent concentric structure comprising red blood cells and calcified fibrinous material (Fig. 3A). Equivocal vascular lumens were observed between phleboliths and muscular fibers, and endothelial cells were completely flattened by compression (Fig. 3B). Nine months postoperatively, the patient developed another painful mass in the right masseter muscle. MRI revealed recurrent hemangioma (Fig. 1B) and reoperation was performed. Under general anesthesia, the incision was made overlapping the scar of the first operation and extended to the tragus of the ear by means of parotidectomy. Postoperative scar formation was gently dissected and the masseter muscle exposed. Following careful exploration, residual phleboliths were completely removed accompanied by fibroadipose tissue (Fig. 4A). Fine vascular connections were observed and completely clamped. Part of the buccal
Fig. 1. (A) Coronal view on preoperative MRI (T2-weighted) showing signal hyperintensity in the right masseter muscle with many round, hypointense areas (arrow). (B) Coronal view on postoperative T2-weighted MRI revealing recurrent hemangioma in right masseter muscle (arrow).
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
Fig. 2. (A) Surgically resected tissue was filled with numerous, round to oval-shaped phleboliths (bar, 1 cm). (B) A total of 21 dark brown phleboliths up to 12 mm in diameter, were observed within the tumor (bar, 1 cm).
branch of the facial nerve was excised to accomplish complete resection of the lesion. To prevent recurrence, normal muscular tissue was also removed well beyond the gross limit (Fig. 4B). Histological examination revealed
589
Fig. 4. (A) Resected phleboliths were accompanied by fibroadipose tissue (bar, 1 cm). (B) After complete removal of recurrent tumor (*parotid gland). Substantial right masseter muscle was resected (arrows).
typical cavernous hemangioma in fibroadipose tissue adjacent to phleboliths, demonstrating that both phleboliths and feeding vessels remained from the previous operation. As of 1 year after reoperation, facial asymmetry had spontaneously improved and no evidence of recurrent disease was seen.
Fig. 3. (A) Histopathology showing prominent phlebolith formation with apparent concentric structure. Lamellated red blood cells and fibrinous materials were seen in phleboliths (10, hematoxylin and eosin (HE)). (B) Equivocal vascular lumens (arrow) were observed between phleboliths (*) and muscular fibers (40, HE).
590
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591
3. Discussion In general, diagnosis of intramuscular hemangioma of the head and neck region can be difficult [4–6]. The lack of objective clinical findings (e.g., compressibility, pulsation, thrills, bruits, evident skin discoloration) and the rare incidence of this lesion allows ample opportunity for misdiagnosis. As only 8% of all cases of intramuscular hemangiomas are diagnosed pre-operatively [5], a variety of parotid and muscular neoplasms, benign muscular hypertrophy, congenital cysts and lymphadenopathies are commonly confused in the differential diagnosis [6]. Phlebolith formation and calcification have been reported as a characteristic feature of hemangiomas [7–9], so diagnostic imaging modalities like CT, MRI [10,11] and ultrasonography have significantly increased the accuracy of preoperative diagnosis for this lesion. Intramuscular hemangiomas are characteristically much brighter on T2- than T1-weighted imaging due to the increased free water present within stagnant blood in the vessels. In this case, prominent phlebolith formation on T2-weighted imaging led to appropriate preoperative diagnosis. Phlebolith formation within intramuscular hemangioma is present in approximately 25% of cases [7,8]. The pathogenesis of phleboliths [12,13] is thought to involve thrombi produced by slowing of peripheral blood flow becoming organized and mineralized. The concentric structure on the cut surface of the phlebolith with histopathology suggests the following growth mechanism. Initially, calcification of the thrombus occurs, forming the core of the phlebolith. The fibrinous component then undergoes secondary calcification and becomes attached. Repetition of this process causes enlargement of the phlebolith. In our patient, long-term presence of hemangioma and stagnant blood flow caused a large number of phleboliths. Many forms of therapy have been advocated to cure or control intramuscular hemangioma [3,11]. Sclerosing agents provide a palliative treatment for intramuscular hemangioma, but post-injection scarring and recurrence have limited their administration. Systemic steroids have been used successfully in treatment of cutaneous capillary hemangiomas [11]. In our case, however, administration of sclerosing agents and/or systemic steroids would not be effective because blood flow in the tumor vessels has already stagnated by prominent phlebolith formation. The ideal treatment is complete excision of the tumor with a surrounding margin of normal muscular tissue, due to the infiltrative nature of the tumor [6,14–16]. Minor feeding vessels and residual tumor are responsible for a recurrence rate of approximately 18%, with 7% recurring more than once [2]. In our experience with this case, a few phleboliths were left in the first operation due to the unnecessary risks of facial palsy. Incomplete removal was followed by recurrences and deeper invasion of adjacent
tissues by tumor. As a result of recurrence, we had to perform reoperation with extended skin incision and deeper dissection in scarred muscular tissue. Phleboliths were certainly observed within the tumor tissue histologically, so careful exploration and complete extirpation of phleboliths is to be strongly recommended even if vascular formations or connecting vessels are not evident near the phleboliths. Excessive blood loss during surgery may result in significant surgical morbidity. Preoperative arterial embolization can be invaluable in diminishing blood loss and is thus widely used during surgery on vascular tumors. However, Ichimura et al. [14] noted that complete tumor resection could be performed with less bleeding if a wide surgical field exposing the whole tumor and surrounding area was obtained. Ligation of feeding vessels also helps to minimize blood loss. In practice, ligating the masseteric branch of the facial artery is sufficient, as most intramuscular hemangiomas including the present case arise in the lower part of the masseter muscle [14,17]. Prominent formation of phleboliths had already decreased blood flow, and ligation of the facial artery in the first operation diminished extensive blood loss in our surgery.
References [1] Barnes L. Tumors and tumor like lesions of the soft tissue. In: Barnes L, editor. Pathology of the head and neck. 2nd ed., New York, NY: Marcel Dekker; 2001. p. 900–1. [2] Allen PW, Enzinger FM. Hemangioma of skeletal muscle. An analysis of 89 cases. Cancer 1972;29:8–22. [3] Wolf GT, Daniel F, Krause CJ, Kaufman RS. Intramuscular hemangioma of the head and neck. Laryngoscope 1985;95:210–3. [4] Elahi MM, Parnes L, Fox A. Hemangioma of the masseter muscle. J Otolaryngol 1992;21:177–9. [5] Avci G, Yim S, Misirliogolu A, Akoz T, Kartal LK. Intramasseteric hemangioma. Plast Reconst Surg 2002;109:1748–9. [6] Demir Z, Oktem F, Celebioglu S. Rare case of intramasseteric cavernous hemangioma in a three-year-old boy: a diagnostic dilemma. Ann Otol Rhinol Laryngol 2004;113:455–8. [7] Morris SJ, Adams H. Case report: paediatric intramuscular haemangiomata—don’t overlook the phlebolith! Br J Radiol 1995;68: 208–11. [8] Osada K, Yoshihara T, Itoh M. Intramasseter hemangiomas: a case report. J Otolaryngol 2000;29:166–9. [9] Mandel L. Intramuscular hemangioma with phleboliths or choristoma? J Oral Maxillofac Surg 2001;59:1511–2. [10] Yonetsu K, Nakayama E, Miwa K, Tanaka T, Araki K, Kanda S, et al. Magnetic resonance imaging of oral and maxillofacial angiomas. Oral Surg Oral Med Oral Pathol 1993;76:783–9. [11] Rossiter JL, Hendrix RA, Tom LWC, Potsic WP. Intramuscular hemangioma of the head and neck. Otolaryngol Head Neck Surg 1993;108:18–26. [12] Sano K, Ogawa A, Inokuchi T, Takahashi H, Hisatsune K, Eng D. Buccal hemangioma with phleboliths; report of two cases. Oral Surg Oral Med Oral Pathol 1988;65:151–6. [13] Raymond AK, Batsakis JG. Pathology consultation: angiolithiasis and sialolithiasis in the head and neck. Ann Otol Rhinol Laryngol 1992;101:455–7.
H. Kanaya et al. / Auris Nasus Larynx 35 (2008) 587–591 [14] Ichimura K, Nibu K, Tanaka T. Essentials of surgical treatment for intramasseteric hemangioma. Eur Arch Otorhinolaryngol 1995;252: 125–9. [15] Welsh D, Hengerer AS. The diagnosis and treatment of intramuscular hemangiomas of the masseter muscle. Am J Otolaryngol 1980;1:186–90.
591
[16] Hart B, Schwartz HC. Cavernous hemangioma of the masseter muscle: report of a case. J Oral Maxillofac Surg 1995;53: 467–9. [17] Ichimura K, Tanaka T, Kitahara N. Surgery of the masseter muscle. Pract Otol (Kyoto) 1989;82:1209–19.