CLINICAL STUDY
Intramuscular Cavernous Hemangioma of the Temporalis Muscle Mehmet Akif Eryilmaz, MD,* Yasin Kürşad Varsak, MD,† Zuhal Gül, MD,* and Ayşenur Uğur, MD* Abstract: Hemangiomas are frequent benign hereditary vascular tumors. Intramuscular hemangiomas, a distinctive type of hemangioma occurring within the skeletal muscle, account for less than 1% of all hemangiomas. They occur more often in the trunk and extremity muscles, whereas the involvement of the temporal muscle is extremely rare. A 34-year-old man with a mass in his left temporal fossa was admitted. Computed tomographic scan showed no erosion of the bone, and magnetic resonance imaging revealed an ovoid mass within the temporal muscle. The lesion was surgically excised, and histopathologic examination confirmed the diagnosis of cavernous hemangioma. The patient was not able to lift his left eyebrow right after the surgery. Two months after the surgery, the patient recovered from paralysis, and there was no recurrence of tumor 12 months after the surgery. We report the 27th cavernous hemangioma case of the temporalis muscle. Care must be taken to avoid possible stretch injury to facial nerve branches while resecting these tumors. Key Words: Benign tumour, hemangioma, intramuscular neoplasms, temporalis muscle (J Craniofac Surg 2014;25: 1400–1401)
H
emangiomas are the most common benign tumors of infancy and childhood. They most often occur in the skin or subcutaneous tissue. Hemangiomas are divided into 3 types according to vessel size: small-vessel (capillary), large-vessel (cavernous), and mixed.1 Less than 1% of all hemangiomas are intramuscular, with the lower limbs being the most frequent localization (45%), followed by the upper limbs (27%) and the trunk (14%). Only 14% are located in the head and neck region, with the masseter, trapezius, and sternocleidomastoideus as the most common sites.2 The temporalis muscle is a very uncommon site within the head and neck region, so there has been only 26 case reports presented in the English literature. Intramuscular hemangioma mostly seen in young adults and no sex preference are noted.3 According to Scott,4 hemangiomas arise in abnormal embryonic sequestrations that retain their embryonic characteristic. Minor trauma or excessive muscle contraction are believed to play a role in the growth of the mass by stimulating blood flow in the preexisting tumor.5 We encountered a patient with a cavernous hemangioma located in the temporalis muscle. From the Departments of Otorhinolaryngology, Necmettin Erbakan University, Meram Medical Faculty; and †Otorhinolaryngology, Konya Numune Hospital, Konya, Turkey. Received January 23, 2014. Accepted for publication February 16, 2014. Address correspondence and reprint requests to Yasin Kürşad Varsak, MD, Department of Otorhinolaryngology, Necmettin Erbakan Universitesi Meram Tip Fakultesi KBB Servisi S Blok Zemin Kat Meram /Konya, Postal Code 42080, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000927
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PATIENT A 34 year-old man was admitted to our clinic, with a 6-month history of a slowly growing painless mass in the left temporal region. The results of his neurologic and physical examinations were normal, except a solid immobile mass lesion approximately 2 cm in diameter in his left temporal fossa. No pulsation or bruit was observed, and the overlying skin was also normal. The patient had been treated for headache at a neurosurgical clinic before he noticed a swelling on his left temporal region. Subsequently, a magnetic resonance imaging (MRI) scan was performed, which revealed a 13 33 18-cm ovoid mass within the left temporal muscle (Fig. 1). The mass was not invading the surrounding tissues. Computed tomographic (CT) scan showed no erosion of the bone and no calcifications. The patient was operated on under general anesthesia. A 3-cm vertical skin incision was placed 1 cm behind the hairline over the scalp in the temporal region. Skin flap was elevated from the underlying temporal fascia; a dark red soft well-demarcated tumor lying in the temporal muscle was encountered. The lesion was excised totally. There was no evidence of infiltration in the surrounding muscle (Fig. 2). Histopathologic examination of the specimen showed large cavernous vascular structures divided by hyalinized fibrous septa, and the diagnosis was cavernous hemangioma (Fig. 3).The patient was not able to lift his left eyebrow right after the surgery. The patient was discharged from the hospital on the third postoperative day. Two months after the surgery, the patient recovered from eyebrow paralysis, so there were no restrictions of eyebrow movements on physical examination and there was no recurrence of tumor.
DISCUSSION Intramuscular hemangiomas are rare tumors accounting for 0.8% of all hemangiomas, and approximately 14% of cases are localized in the musculature of the head and neck region.6,7 The masseter (36%), followed by the trapezius (24%), are the muscles most frequently affected.6 However, hemangioma in the temporalis muscle is rarely reported in the literature. To the best of our knowledge, in the international literature, Bucci et al8 reported a review of temporal muscle hemangiomas, which has shown that 24 cases exist in the literature as of 2008. Another case was reported by Çalışaneller et al9 before this review. Recently, Gadhia et al10 reported a cavernous haemangioma in the temporalis muscle with multiple phleboliths in 2011. Our patient is the 27th case in the literature. Intramuscular hemangiomas were classified, in 1972, by Allen and Enzinger1 depending on the vessel size. Histologically, intramuscular hemangiomas are classified into several categories including capillary hemangioma, cavernous hemangioma, and venous hemangioma. Capillary hemangioma is composed of small capillary–sized vessels, whereas cavernous hemangioma consists of thin-walled and cystic blood spaces. Venous hemangioma is composed of dilated veins with walls of varying thickness within loose fatty-fibrous stromas.11 In our patient, pathologic findings revealed a cavernous hemangioma that was identified through cavernous vascular structures divided by hyalinized fibrous septa. Hemangiomas are readily distinguished from other soft-tissue tumors through CT, MRI, and arteriography.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Temporalis Muscle Cavernous Hemangioma
FIGURE 3. Histopathologic image shows cavernous vascular structures divided by hyalinized fibrous septa. FIGURE 1. T1-weighted coronal MRI showing the 13 33 18-cm ovoid mass within the left temporal muscle.
Computed tomography is useful for defining the form, size, and anatomic relationship of the tumor, but MRI is the method of choice in defining the vascular nature of the tumor. On T1-weighted imaging, hemangiomas are isointense or hypointense to muscle. With T2 imaging, the lesions are hyperintense on account of the volume of stagnant blood, clearly differentiated from the normal muscle and fibrofatty septa.12 Certain radiologic features, on MRI, would appear to suggest hemangiomas: 1) high-intensity signal on T2-weighted imaging; 2) endothelial-lined vascular channels separated by fibrous and/or fatty linear tissue in lesions bigger than 2 cm; 3) Presence of areas of thrombus, fibrosis, hemosiderin deposition, and/or calcification12 Arteriography is generally used for preoperative embolization. However, our patient’s tumor was not that large to require an embolization. Aspiration cytology can point to the vascular origin of the tumor but may not disclose its exact nature. Vascular channels in the tumor often produce a bloody sample difficult to read. Open biopsy is rarely contemplated because of the danger of bleeding.5 The treatment options of intramuscular hemangioma in the temporal muscle should be individualized with respect to localization, depth of invasion, vascularization, and clinical symptoms, which are simple observation, irradiation, injection of sclerosing agents, corticosteroid treatment, embolization, and surgical excision. Irradiation is not recommended because the amount of radiation needed is very high and has severe potential complications especially in children.13 Only 1 venous hemangioma in the temporal muscle that disappeared spontaneously has been reported in the literature.14 We treated our patient surgically under general anesthesia. Vertical skin incision is used
FIGURE 2. Intraoperative view of the hemangioma.
to secure the temporal artery and vein. Incision was placed behind the hairline over the scalp to hide the incision scar so that the cosmetic results will be excellent. Careful surgical dissection during excision of a temporal muscle hemangioma is important to prevent injury to the temporal and auricular branches of the facial nerve. Unfortunately, a complication that caused him to not be able to lift his left eyebrow right after the surgery was observed in our patient. The patient recovered 2 months after the surgery, so we thought that surgical manipulation was responsible for the paralyses. Local recurrence after surgery has been attributed to incomplete excision and not to the histopathologic type.11 There was no recurrence in our patient 6 months after the surgery. In conclusion, cavernous hemangiomas should be suspected when swelling occurs in the temporal region accompanying radiologic findings, which suggests vascular origin. Care should be taken to avoid facial nerve injury even for smaller lesions.
REFERENCES 1. Allen PW, Enzinger FW. Hemangioma of skeletal muscle: an analysis of 89 cases. Cancer 1972;29:8–22 2. Batsakis JG. Tumors of the head and neck: clinical and pathological considerations. Baltimore: Williams & Wilkins, 1979:249–296 3. Enzinger FM, Weiss SW. Soft tissue tumors. St Louis: CV Mosby, 1988:512–514 4. Scott JES. Hemangiomata in skeletal muscle. Br J Surg 1957;44:496–501 5. Fergusson ILC. Hemangiomata of skeletal muscle. Br J Surg 1972;59:634–637 6. Odabasi AO, Metin KK, Mutlu C, et al. Intramuscular hemangioma of the masseter muscle. Eur Arch Otorhinolaryngol 1999;256:366–369 7. Shpitzer T, Noyek AM, Witterick I, et al. Noncutaneous cavernous hemangiomas of the head and neck. Am J Otolaryngol 1997;18:367–374 8. Bucci T, De Giulio F, Romano A, et al. Cavernous haemangioma of the temporalis: case report and review of the literature Acta Otorhinolaryngol Ital 2008;28:83–86 9. Çalışaneller T, Özdemir Ö, Yıldırım E, et al. Cavernous hemangioma of temporalis muscle: report of a case and review of the literature. Turk Neurosurg 2007;17:33–36 10. Gadhia K, Bunyan R, Chan CH. Multiple radio-opacities in an OPG: a case report of cavernous haemangioma of temporalis muscle with multiple phleboliths. Dent Update 2011;38:711–713 11. Beham A, Fletcher CD. Intramuscular angioma: a clinicopathological analysis of 74 cases. Histopathology 1991;18:53–59 12. Buetow PC, Kransdorf MJ, Moser RP, et al. Radiologic appearance of intramuscular hemangioma with emphasis on MR imaging. AJR Am J Roentgenol 1990;154:5637 13. Bartlett JA, Riding KH, Salkeld LJ. Management of hemangiomas of the head and neck in children. J Otolaryngol 1988;17:111–120 14. Itosaka H, Tada M, Sawamura Y, et al. Vanishing tumor of the temporalis muscle: repeated hemorrhage in an intramuscular venous hemangioma. AJNR Am J Neuroradiol 1997;18:983–985
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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Intramuscular Cavernous Hemangioma of the Temporalis Muscle Mehmet Akif Eryilmaz, MD,* Yasin Kürşad Varsak, MD,† Zuhal Gül, MD,* and Ayşenur Uğur, MD* Abstract: Hemangiomas are frequent benign hereditary vascular tumors. Intramuscular hemangiomas, a distinctive type of hemangioma occurring within the skeletal muscle, account for less than 1% of all hemangiomas. They occur more often in the trunk and extremity muscles, whereas the involvement of the temporal muscle is extremely rare. A 34-year-old man with a mass in his left temporal fossa was admitted. Computed tomographic scan showed no erosion of the bone, and magnetic resonance imaging revealed an ovoid mass within the temporal muscle. The lesion was surgically excised, and histopathologic examination confirmed the diagnosis of cavernous hemangioma. The patient was not able to lift his left eyebrow right after the surgery. Two months after the surgery, the patient recovered from paralysis, and there was no recurrence of tumor 12 months after the surgery. We report the 27th cavernous hemangioma case of the temporalis muscle. Care must be taken to avoid possible stretch injury to facial nerve branches while resecting these tumors. Key Words: Benign tumour, hemangioma, intramuscular neoplasms, temporalis muscle (J Craniofac Surg 2014;25: 1400–1401)
H
emangiomas are the most common benign tumors of infancy and childhood. They most often occur in the skin or subcutaneous tissue. Hemangiomas are divided into 3 types according to vessel size: small-vessel (capillary), large-vessel (cavernous), and mixed.1 Less than 1% of all hemangiomas are intramuscular, with the lower limbs being the most frequent localization (45%), followed by the upper limbs (27%) and the trunk (14%). Only 14% are located in the head and neck region, with the masseter, trapezius, and sternocleidomastoideus as the most common sites.2 The temporalis muscle is a very uncommon site within the head and neck region, so there has been only 26 case reports presented in the English literature. Intramuscular hemangioma mostly seen in young adults and no sex preference are noted.3 According to Scott,4 hemangiomas arise in abnormal embryonic sequestrations that retain their embryonic characteristic. Minor trauma or excessive muscle contraction are believed to play a role in the growth of the mass by stimulating blood flow in the preexisting tumor.5 We encountered a patient with a cavernous hemangioma located in the temporalis muscle. From the Departments of Otorhinolaryngology, Necmettin Erbakan University, Meram Medical Faculty; and †Otorhinolaryngology, Konya Numune Hospital, Konya, Turkey. Received January 23, 2014. Accepted for publication February 16, 2014. Address correspondence and reprint requests to Yasin Kürşad Varsak, MD, Department of Otorhinolaryngology, Necmettin Erbakan Universitesi Meram Tip Fakultesi KBB Servisi S Blok Zemin Kat Meram /Konya, Postal Code 42080, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000927
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PATIENT A 34 year-old man was admitted to our clinic, with a 6-month history of a slowly growing painless mass in the left temporal region. The results of his neurologic and physical examinations were normal, except a solid immobile mass lesion approximately 2 cm in diameter in his left temporal fossa. No pulsation or bruit was observed, and the overlying skin was also normal. The patient had been treated for headache at a neurosurgical clinic before he noticed a swelling on his left temporal region. Subsequently, a magnetic resonance imaging (MRI) scan was performed, which revealed a 13 33 18-cm ovoid mass within the left temporal muscle (Fig. 1). The mass was not invading the surrounding tissues. Computed tomographic (CT) scan showed no erosion of the bone and no calcifications. The patient was operated on under general anesthesia. A 3-cm vertical skin incision was placed 1 cm behind the hairline over the scalp in the temporal region. Skin flap was elevated from the underlying temporal fascia; a dark red soft well-demarcated tumor lying in the temporal muscle was encountered. The lesion was excised totally. There was no evidence of infiltration in the surrounding muscle (Fig. 2). Histopathologic examination of the specimen showed large cavernous vascular structures divided by hyalinized fibrous septa, and the diagnosis was cavernous hemangioma (Fig. 3).The patient was not able to lift his left eyebrow right after the surgery. The patient was discharged from the hospital on the third postoperative day. Two months after the surgery, the patient recovered from eyebrow paralysis, so there were no restrictions of eyebrow movements on physical examination and there was no recurrence of tumor.
DISCUSSION Intramuscular hemangiomas are rare tumors accounting for 0.8% of all hemangiomas, and approximately 14% of cases are localized in the musculature of the head and neck region.6,7 The masseter (36%), followed by the trapezius (24%), are the muscles most frequently affected.6 However, hemangioma in the temporalis muscle is rarely reported in the literature. To the best of our knowledge, in the international literature, Bucci et al8 reported a review of temporal muscle hemangiomas, which has shown that 24 cases exist in the literature as of 2008. Another case was reported by Çalışaneller et al9 before this review. Recently, Gadhia et al10 reported a cavernous haemangioma in the temporalis muscle with multiple phleboliths in 2011. Our patient is the 27th case in the literature. Intramuscular hemangiomas were classified, in 1972, by Allen and Enzinger1 depending on the vessel size. Histologically, intramuscular hemangiomas are classified into several categories including capillary hemangioma, cavernous hemangioma, and venous hemangioma. Capillary hemangioma is composed of small capillary–sized vessels, whereas cavernous hemangioma consists of thin-walled and cystic blood spaces. Venous hemangioma is composed of dilated veins with walls of varying thickness within loose fatty-fibrous stromas.11 In our patient, pathologic findings revealed a cavernous hemangioma that was identified through cavernous vascular structures divided by hyalinized fibrous septa. Hemangiomas are readily distinguished from other soft-tissue tumors through CT, MRI, and arteriography.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Temporalis Muscle Cavernous Hemangioma
FIGURE 3. Histopathologic image shows cavernous vascular structures divided by hyalinized fibrous septa. FIGURE 1. T1-weighted coronal MRI showing the 13 33 18-cm ovoid mass within the left temporal muscle.
Computed tomography is useful for defining the form, size, and anatomic relationship of the tumor, but MRI is the method of choice in defining the vascular nature of the tumor. On T1-weighted imaging, hemangiomas are isointense or hypointense to muscle. With T2 imaging, the lesions are hyperintense on account of the volume of stagnant blood, clearly differentiated from the normal muscle and fibrofatty septa.12 Certain radiologic features, on MRI, would appear to suggest hemangiomas: 1) high-intensity signal on T2-weighted imaging; 2) endothelial-lined vascular channels separated by fibrous and/or fatty linear tissue in lesions bigger than 2 cm; 3) Presence of areas of thrombus, fibrosis, hemosiderin deposition, and/or calcification12 Arteriography is generally used for preoperative embolization. However, our patient’s tumor was not that large to require an embolization. Aspiration cytology can point to the vascular origin of the tumor but may not disclose its exact nature. Vascular channels in the tumor often produce a bloody sample difficult to read. Open biopsy is rarely contemplated because of the danger of bleeding.5 The treatment options of intramuscular hemangioma in the temporal muscle should be individualized with respect to localization, depth of invasion, vascularization, and clinical symptoms, which are simple observation, irradiation, injection of sclerosing agents, corticosteroid treatment, embolization, and surgical excision. Irradiation is not recommended because the amount of radiation needed is very high and has severe potential complications especially in children.13 Only 1 venous hemangioma in the temporal muscle that disappeared spontaneously has been reported in the literature.14 We treated our patient surgically under general anesthesia. Vertical skin incision is used
FIGURE 2. Intraoperative view of the hemangioma.
to secure the temporal artery and vein. Incision was placed behind the hairline over the scalp to hide the incision scar so that the cosmetic results will be excellent. Careful surgical dissection during excision of a temporal muscle hemangioma is important to prevent injury to the temporal and auricular branches of the facial nerve. Unfortunately, a complication that caused him to not be able to lift his left eyebrow right after the surgery was observed in our patient. The patient recovered 2 months after the surgery, so we thought that surgical manipulation was responsible for the paralyses. Local recurrence after surgery has been attributed to incomplete excision and not to the histopathologic type.11 There was no recurrence in our patient 6 months after the surgery. In conclusion, cavernous hemangiomas should be suspected when swelling occurs in the temporal region accompanying radiologic findings, which suggests vascular origin. Care should be taken to avoid facial nerve injury even for smaller lesions.
REFERENCES 1. Allen PW, Enzinger FW. Hemangioma of skeletal muscle: an analysis of 89 cases. Cancer 1972;29:8–22 2. Batsakis JG. Tumors of the head and neck: clinical and pathological considerations. Baltimore: Williams & Wilkins, 1979:249–296 3. Enzinger FM, Weiss SW. Soft tissue tumors. St Louis: CV Mosby, 1988:512–514 4. Scott JES. Hemangiomata in skeletal muscle. Br J Surg 1957;44:496–501 5. Fergusson ILC. Hemangiomata of skeletal muscle. Br J Surg 1972;59:634–637 6. Odabasi AO, Metin KK, Mutlu C, et al. Intramuscular hemangioma of the masseter muscle. Eur Arch Otorhinolaryngol 1999;256:366–369 7. Shpitzer T, Noyek AM, Witterick I, et al. Noncutaneous cavernous hemangiomas of the head and neck. Am J Otolaryngol 1997;18:367–374 8. Bucci T, De Giulio F, Romano A, et al. Cavernous haemangioma of the temporalis: case report and review of the literature Acta Otorhinolaryngol Ital 2008;28:83–86 9. Çalışaneller T, Özdemir Ö, Yıldırım E, et al. Cavernous hemangioma of temporalis muscle: report of a case and review of the literature. Turk Neurosurg 2007;17:33–36 10. Gadhia K, Bunyan R, Chan CH. Multiple radio-opacities in an OPG: a case report of cavernous haemangioma of temporalis muscle with multiple phleboliths. Dent Update 2011;38:711–713 11. Beham A, Fletcher CD. Intramuscular angioma: a clinicopathological analysis of 74 cases. Histopathology 1991;18:53–59 12. Buetow PC, Kransdorf MJ, Moser RP, et al. Radiologic appearance of intramuscular hemangioma with emphasis on MR imaging. AJR Am J Roentgenol 1990;154:5637 13. Bartlett JA, Riding KH, Salkeld LJ. Management of hemangiomas of the head and neck in children. J Otolaryngol 1988;17:111–120 14. Itosaka H, Tada M, Sawamura Y, et al. Vanishing tumor of the temporalis muscle: repeated hemorrhage in an intramuscular venous hemangioma. AJNR Am J Neuroradiol 1997;18:983–985
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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