Accepted Manuscript Masseteric intramuscular hemangioma: case report• Jason Gordon, BA Louis Mandel, DDS PII:
S0278-2391(14)00584-9
DOI:
10.1016/j.joms.2014.06.001
Reference:
YJOMS 56351
To appear in:
Journal of Oral and Maxillofacial Surgery
Received Date: 9 May 2014 Revised Date:
2 June 2014
Accepted Date: 3 June 2014
Please cite this article as: Gordon J, Mandel L, Masseteric intramuscular hemangioma: case report•, Journal of Oral and Maxillofacial Surgery (2014), doi: 10.1016/j.joms.2014.06.001. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
MASSETERIC INTRAMUSCULAR HEMANGIOMA: CASE REPORT•
SC
RI PT
Jason Gordon BA•• Louis Mandel DDS•••
•From the Columbia University College of Dental Medicine Salivary Gland Center, New York, NY
M AN U
••Research Assistant, Salivary Gland Center; Third Year Student, College of Dental Medicine
TE D
•••Director, Salivary Gland Center; Associate Dean, Clinical Professor (Oral and Maxillofacial Surgery), Columbia University College of Dental Medicine 630 West 168th Street, New York NY, 10032
AC C
EP
Corresponding Author: Louis Mandel DDS College of Dental Medicine 630 West 168th Street New York, NY 10032
Phone: 212 305-9982 Fax: 212 305-9659 Email: [email protected]
ACCEPTED MANUSCRIPT
MASSETERIC INTRAMUSCULAR HEMANGIOMA: CASE REPORT
SC
RI PT
Jason Gordon BA•• Louis Mandel DDS•••
•From the Columbia University College of Dental Medicine Salivary Gland Center, New York, NY
M AN U
••Research Assistant, Salivary Gland Center; Third Year Student, College of Dental Medicine
TE D
•••Director, Salivary Gland Center; Associate Dean, Clinical Professor (Oral and Maxillofacial Surgery), Columbia University College of Dental Medicine 630 West 168th Street, New York NY, 10032
AC C
EP
Corresponding Author: Louis Mandel DDS College of Dental Medicine 630 West 168th Street New York, NY 10032
Phone: 212 305-9982 Fax: 212 305-9659 Email: [email protected]
ACCEPTED MANUSCRIPT
ABSTRACT
RI PT
Securing a clinical diagnosis for the masseteric intramuscular hemangioma is difficult. However, making a diagnosis can be facilitated with imaging, particularly the magnetic imaging,
which
serves
as
a
key
element
in
preoperative
AC C
EP
TE D
M AN U
SC
resonance
2
diagnosis.
ACCEPTED MANUSCRIPT
The intramuscular hemangioma (IMH), a relatively rare vascular lesion involving skeletal muscle, represents only 1% of all vascular anomalies (1). More commonly seen in the pelvic
the masseter muscle (1-4).
RI PT
area, the head and neck region is where 10-15% of the IMHs occur, with 36% of these involving The IMH is considered a congenital and benign vascular
malformation (VM) that results from an error in blood vessel morphogenesis (5-8). Although
SC
not readily apparent, it is present at birth, grows proportionately with the patient, becomes
M AN U
cosmetically visible in early adult life and demonstrates no sex predilection (5-11).
Because it is embedded within muscle tissue, diagnosing the masseteric IMH has proven to be difficult. There are no objective findings such as a bruit or pulsations, and its depth within the masseter prevents skin discoloration. As a rule there also are no subjective symptoms, other
TE D
than a gradual increase in size such that a cosmetic asymmetry develops, and serves as the impetus for consultation. The swelling can be present for many years, but it becomes a cosmetic issue as the patient enters adulthood. Palpation usually reveals the swelling to be soft, painless,
EP
compressible and not circumscribed. Pain only is an issue when there is rapid growth of the
AC C
masseteric IMH with impingement upon adjacent structures (4, 12).
The diagnosis of a head/neck IMH can be facilitated by the presence of the wattle sign (13) which develops because the VM accumulates blood when the head is prone or in a dependent position. Venous return from the vascular lesion to the superior vena cava is impaired by gravity and an increased swelling from vascular engorgement occurs. Patients often note that the swelling is most marked upon awakening after sleep. With the patient’s return to a vertical
3
ACCEPTED MANUSCRIPT
position, the enlargement gradually dissipates as gravity takes effect. The term “wattle sign” is derived from the enlargement of the male turkey’s wattle as it becomes engorged with blood and
RI PT
functions to attract a female.
Imaging has proven to be a very significant tool in diagnosing the masseteric IMH, particularly if a phlebolith can be identified. When recognized in the presence of the somewhat
SC
limited clinical signs associated with the IMH, the phlebolith plays a key role in pointing to a diagnosis. The varied presence of a phlebolith (3-50% of IMHs) has been reported as an
M AN U
incidental imaging finding (1, 4, 12-15). Vessels subjected to abrupt stretching and rises in pressure during physical activity, such as mastication and exercise, can suffer damage to their intima (16). As part of the healing process, a protective thrombus develops at the point of injury. Alternatively, blood flowing through the tortuous vessels of a vascular lesion, tends to stagnate
TE D
and thrombose. The phlebolith develops when dystrophic calcification occurs within the thrombus. The phlebolith usually has a concentric lamellated pattern, a core that can be lucent or opaque, and consists of a mixture of calcium carbonate and calcium phosphate salts (17, 18).
EP
Small phleboliths tend to be uniformly opaque (19).
AC C
Standard panoramic radiography, in an office setting, can be helpful in uncovering the existence of a IMH phlebolith. However, the CT is the diagnostic approach of choice for phlebolith diagnosis. The scan derives its diagnostic ability from the fact that it is exquisitely sensitive to minute amounts of calcium which it can locate in three dimensions. Contrast should be avoided because its presence in a small vessel mimics the appearance of a phlebolith. Regardless, the CT scan is not the best imaging technique to uncover the presence of an IMH
4
ACCEPTED MANUSCRIPT
because even with contrast the vascularity of the IMH often is not adequately enhanced and phleboliths may be absent. However, some linear or spotty enhancement, reflecting vascularity, may be observed (20). Nevertheless, the CT scan is not sufficient to reliably clinch the presence
RI PT
and extent of vascularity or to demonstrate delineation between the IMH and muscle (10). Because only minimal enhancement is noted, the CT scan cannot convincingly suggest a
SC
diagnosis of IMH without the presence of a phlebolith.
The MRI with gadolinium has proven to be the diagnostic imaging procedure of choice
M AN U
for the IMH. It serves to accurately clarify the existence and location of the IMH within the masseter muscle, the IMH’s relation to adjacent anatomic structures, and when indicated determines the surgical approach. The IMH is isointense, similar to muscle, on a T1-weighted view. However, it enhances clearly, and is hyperintense on a T2-weighted study due to the free
TE D
water content of the vascularity present in the IMH. Low signals in the IMH are also seen in the T2-weighted film, and represent fibrous septae amid the vascular elements (21-25).
EP
The authors report a patient with a masseteric IMH who, other than a mild facial asymmetry, was subjectively asymptomatic. This report illustrates and emphasizes the central
AC C
role that imaging plays in the diagnosis of a masseteric IMH.
CASE REPORT
A 45-year-old female, in excellent health, was referred to the Columbia University Salivary Gland Center (SGC) because of painless swelling in the left parotid/masseteric region (Fig. 1). The patient stated that she has been aware of the facial swelling since the age of 4
5
ACCEPTED MANUSCRIPT
years. No treatment had been sought in the past because there were no subjective symptoms. The patient developed concerns regarding its persistent presence and the associated cosmetic
RI PT
asymmetry.
A CT scan had been previously requested by the patient’s physician (Fig.2). The report indicated the presence of an enlargement of the left masseter muscle.
A calcific density,
SC
measuring .5 x .4cm, was seen in the anterior medial aspect of the left masseter muscle. Abnormal enhancement within the masseter was also observed and thought to represent
M AN U
“postinflammatory/postinfectious change”. Because of the patient’s ensuing apprehension, she decided to seek further consultation in the SGC.
Clinically, a moderate left facial swelling was noted. The patient stated that the swelling
TE D
did not fluctuate in size or cause pain when eating in all the years of its known existence. She did state that the swelling seemed to be larger in the morning upon awakening from sleep, but would subside as the day progressed (the wattle sign). The swelling seemed to irregularly follow It was mostly anterior and inferior to the
EP
the anatomic contour of the masseter muscle.
superficial lobe of the parotid gland. Clenching accentuated the facial asymmetry. Palpation
AC C
indicated the enlargement was soft, painless and not circumscribed. The overlying skin and subcutaneous tissues were movable and there was no surface discoloration.
No cervical
lymphadenopathy was present.
Intraorally, the mucosa was moist. External massage of the left parotid salivary gland produced a normal clear salivary volume from a normal appearing left parotid duct orifice. No swelling or discoloration of the oral buccal mucosa were evident. A panoramic radiograph 6
ACCEPTED MANUSCRIPT
confirmed the presence of the calcific body previously identified by the CT scan (Fig. 3). It was located superimposed upon the anterior aspect of the left mandibular ramus, at the level of the
RI PT
occlusal plane of the mandibular molars.
A MRI with contrast was ordered for further evaluation and diagnosis (Fig. 4). The T2weighted MRI study clearly enhanced a vascular lesion within the left masseter muscle, and
SC
confirmed the presence and location of the phlebolith. A confident diagnosis of an IMH was now indicated. With the patient’s concerns regarding the existence of a neoplasm allayed, definitive
M AN U
therapeutic care was emphatically declined. She did state that she would be willing to return periodically for observation.
DISCUSSION
TE D
The means of diagnosing a masseteric IMH can not be achieved with any certainty from its location, duration, and presence of an asymptomatic swelling. In our patient, a convincing diagnosis could not be reached without the imaging studies. The CT scan identified the presence
EP
of enlargement and enhancement of the masseter, but the origin could not be determined. Clues were obtained from the presence of the calcified body noted on the CT scan and confirmed by
AC C
the panoramic radiograph. When this information was combined the lesion’s history, clinical examination and the T2-weighted MRI finding of a significantly enhanced masseter muscle from contained vascular fluid, a diagnosis of a vascular IMH was made possible.
Phleboliths are very uncommon in the oral region, and their presence suggests a vascular lesion. They can occur in the absence of such lesions, but they are mostly associated with vascular abnormalities. Our patient did not demonstrate the characteristic lamellated feature of a 7
ACCEPTED MANUSCRIPT
phlebolith. However, lamellation may be absent, as in our patient, in smaller phleboliths (19). Phleboliths are not to be confused with sialoliths. Their close proximity to the parotid gland leads to misdiagnosis. Differentiation rests in the absence of parotid sialadenitis and obstructive
RI PT
duct symptomatology. In comparison to sialoliths, phleboliths are usually multiple, more randomly located and lamellated. Furthermore, the ovality of the phlebolith can serve to distinguish it from the sialolith which is frequently elliptically shaped by the contour of the
SC
salivary duct. The IMH phlebolith must also be differentiated from other head and neck calicifications. The calcified lymph node often represents the end product of a lymphadenitis.
M AN U
Calcified acne lesions, atherosclerotic calcifications, residual phleboliths from involuted childhood hemangiomas, tonsilloliths, cysticercosis, and angioliths seen in some syndromes all must be considered.
TE D
The MRI represents the best imaging modality for diagnosis of the IMH. A T2-weighted film will show the IMH to have an intense signal while the surrounding tissues demonstrate low signal intensity. The T2 hyperintensity is due to the free water present in stagnant blood. The
EP
MRI also avoids ionizing radiation and demonstrates excellent contrast between a vascular lesion and adjacent tissues. However, phleboliths are not easily recognized on the T2-weighted film
AC C
because of their very low signal intensity. They are best identified on plain radiography and/or the CT scan.
8
ACCEPTED MANUSCRIPT
ILLUSTRATIONS
Clinical view of left facial swelling
Fig. 2.
CT scan (with contrast) demonstrates enlarged left masseter muscle
RI PT
Fig. 1.
containing phlebolith (black arrow). Contrast dye highlights vascular
Panoramic radiograph reveals phlebolith superimposed upon anterior
M AN U
Fig. 3.
SC
tissue (white arrow)
aspect of left mandibular ramus (arrow).
MRI with contrast (T2-weighted). Note hyperintense area involving left masseter muscle caused by IMH vascularity (arrowheads). Phlebolith
EP
TE D
(hypointense nodule) present (arrow).
AC C
Fig. 4.
9
ACCEPTED MANUSCRIPT
REFERENCE
1. Zengin AZ, Celenk P, Sumer AP: Intramuscular hemangioma presenting with multiple
RI PT
phleboliths. Oral Surg Oral Med Oral Pathol Oral Radiol 115: 32e, 2013
2. Yang WT, Ahuja A, Metreweli C: Sonographic features of head and neck hemangiomas
SC
and vascular malformations. A review of 23 patients. J Ultrasound Med 16: 39, 1997
M AN U
3. Avci G, Yim S, Misiriogolu A, et al: Intramasseteric hemangioma. Plast Reconstr Surg 109: 1748, 2002
4. Capote A, Acero J, Garcia-Recuero I, Rey J, Guerra B, de Paz V: Infratemporalpreauricular-cervical approach for resection of a cavernous intramasseteric hemangioma:
TE D
A case report. J Oral Maxillofac Surg 66: 2393, 2008
5. Mulliken JB, Glowacki J: Hemangiomas and vascular malformations in infants and
AC C
412, 1982
EP
children: a classification based on endothelial characteristics. Plast Reconstr Surg. 69:
6. Kaban L, Mulliken JB: Vascular anomalies of the maxillofacial region. J Oral Maxillofac Surg 44: 203, 1986
7. Mulliken JB, Fishman SJ, Burrows PE: Vascular anomalies. Curr Probl Surg. 37: 517, 2000
10
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8. Scolozzi P, Laurent F, Lombardi T, Richter M: Intraoral venous malformation presenting with multiple phleboliths. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 96; 197,
RI PT
2003
9. Odabasi AO, Metin KK, Mutlu C, et al: Intramuscular hemangioma of the masseteric muscle. Eur Arch Otorhinolaryngel 256:366, 1999
SC
10. Kakimoto N, Tanimoto K, Nishiyama H, Murakami S, Furukawa S, Kreiborg S. CT and MR imaging of oral and maxillofacial hemangioma and vascular malformation. Eur J
M AN U
Radiol 55: 108, 2005
11. Altug HA, Buyuksoy V, Okcu KM, Dogan N: Hemangiomas of the head and neck with phleboliths: Clinical features, diagnostic imaging, and treatment of 3 cases. Oral Surg
TE D
Oral Med Oral Pathol Oral Radiol Endod 103: 60e, 2007
12. Rossiter JL, Hendrix RA, Tom LWC, et al: Intramuscular hemangioma of the head and
EP
neck. Head Neck Surg 108: 18, 1993
AC C
13. Mandel L, Surattanont F: Clinical and imaging diagnoses of intramuscular hemangiomas: the wattle sign and case reports. J Oral Maxillofac Surg 62: 754, 2004
14. Elahi M, Parnes L, Fox A: Hemangioma of the masseter muscle. J Otolaryngol 21: 177, 1992
11
ACCEPTED MANUSCRIPT
15. Morris SJ, Adams H: Case report: Pediatric intramuscular hemangiomata-Don’t overlook the phlebolith! BR J Radiol 68: 208, 1995
17. Ribbert H: Die phlebolithen. Virchows Arch 223: 339, 1917
RI PT
16. Shemilt P: The origin of phleboliths. Br J Surg 59: 695, 1972
SC
18. Sano K, Ogawa A, Inokuch T, et al: Buccal hemangioma with phlebolith. Oral Surg
M AN U
Oral Med Oral Pathol 65: 151, 1988
19. Kato H, Ota Y, Sasaki M, Arai T, Sekido Y, Tsukinoki K: A phlebolith in the anterior portion of the massetyer muscle. Tokai J Exp Clin Med 37: 25, 2012
20. Lee SK, Kwon SY: Intramuscular cavernous hemangioma arising from masseter muscle:
TE D
a diagnostic dilemma. Eur Radiol 17: 854, 2007
21. Itoh K, Nashimura KT, Togashi K et al: MR imaging of cavernous hemangioma of the
EP
face and neck. J Comput Assist Tomogr 10: 831, 1986
22. Kaplan PA, William SM: Mucocutaneous and peripheral soft-tissue hemangiomas: MR
AC C
imaging. Radiology 163: 163, 1987
23. Mandelblatt SM, Braun IF, Davis PC, et al: Parotid masses: MR imaging. Radiology 163: 411, 1987
12
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24. Jani P, Robb P, Hanby AM, et al: Intramuscular hemangioma of the sternocleidomastoid muscle. Head Neck 12: 164, 1990
RI PT
25. Buetow PC, Kransdorf MJ, Moser RP, et al: Radiologic imaging of intramuscular
AC C
EP
TE D
M AN U
SC
hemangiomas with emphasis on MR imaging. AJR Am J Roentgenol 154: 563, 1990
13
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EP
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SC
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EP
TE D
M AN U
SC
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ACCEPTED MANUSCRIPT
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EP
TE D
M AN U
SC
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ACCEPTED MANUSCRIPT
S0278-2391(14)00584-9
DOI:
10.1016/j.joms.2014.06.001
Reference:
YJOMS 56351
To appear in:
Journal of Oral and Maxillofacial Surgery
Received Date: 9 May 2014 Revised Date:
2 June 2014
Accepted Date: 3 June 2014
Please cite this article as: Gordon J, Mandel L, Masseteric intramuscular hemangioma: case report•, Journal of Oral and Maxillofacial Surgery (2014), doi: 10.1016/j.joms.2014.06.001. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
MASSETERIC INTRAMUSCULAR HEMANGIOMA: CASE REPORT•
SC
RI PT
Jason Gordon BA•• Louis Mandel DDS•••
•From the Columbia University College of Dental Medicine Salivary Gland Center, New York, NY
M AN U
••Research Assistant, Salivary Gland Center; Third Year Student, College of Dental Medicine
TE D
•••Director, Salivary Gland Center; Associate Dean, Clinical Professor (Oral and Maxillofacial Surgery), Columbia University College of Dental Medicine 630 West 168th Street, New York NY, 10032
AC C
EP
Corresponding Author: Louis Mandel DDS College of Dental Medicine 630 West 168th Street New York, NY 10032
Phone: 212 305-9982 Fax: 212 305-9659 Email: [email protected]
ACCEPTED MANUSCRIPT
MASSETERIC INTRAMUSCULAR HEMANGIOMA: CASE REPORT
SC
RI PT
Jason Gordon BA•• Louis Mandel DDS•••
•From the Columbia University College of Dental Medicine Salivary Gland Center, New York, NY
M AN U
••Research Assistant, Salivary Gland Center; Third Year Student, College of Dental Medicine
TE D
•••Director, Salivary Gland Center; Associate Dean, Clinical Professor (Oral and Maxillofacial Surgery), Columbia University College of Dental Medicine 630 West 168th Street, New York NY, 10032
AC C
EP
Corresponding Author: Louis Mandel DDS College of Dental Medicine 630 West 168th Street New York, NY 10032
Phone: 212 305-9982 Fax: 212 305-9659 Email: [email protected]
ACCEPTED MANUSCRIPT
ABSTRACT
RI PT
Securing a clinical diagnosis for the masseteric intramuscular hemangioma is difficult. However, making a diagnosis can be facilitated with imaging, particularly the magnetic imaging,
which
serves
as
a
key
element
in
preoperative
AC C
EP
TE D
M AN U
SC
resonance
2
diagnosis.
ACCEPTED MANUSCRIPT
The intramuscular hemangioma (IMH), a relatively rare vascular lesion involving skeletal muscle, represents only 1% of all vascular anomalies (1). More commonly seen in the pelvic
the masseter muscle (1-4).
RI PT
area, the head and neck region is where 10-15% of the IMHs occur, with 36% of these involving The IMH is considered a congenital and benign vascular
malformation (VM) that results from an error in blood vessel morphogenesis (5-8). Although
SC
not readily apparent, it is present at birth, grows proportionately with the patient, becomes
M AN U
cosmetically visible in early adult life and demonstrates no sex predilection (5-11).
Because it is embedded within muscle tissue, diagnosing the masseteric IMH has proven to be difficult. There are no objective findings such as a bruit or pulsations, and its depth within the masseter prevents skin discoloration. As a rule there also are no subjective symptoms, other
TE D
than a gradual increase in size such that a cosmetic asymmetry develops, and serves as the impetus for consultation. The swelling can be present for many years, but it becomes a cosmetic issue as the patient enters adulthood. Palpation usually reveals the swelling to be soft, painless,
EP
compressible and not circumscribed. Pain only is an issue when there is rapid growth of the
AC C
masseteric IMH with impingement upon adjacent structures (4, 12).
The diagnosis of a head/neck IMH can be facilitated by the presence of the wattle sign (13) which develops because the VM accumulates blood when the head is prone or in a dependent position. Venous return from the vascular lesion to the superior vena cava is impaired by gravity and an increased swelling from vascular engorgement occurs. Patients often note that the swelling is most marked upon awakening after sleep. With the patient’s return to a vertical
3
ACCEPTED MANUSCRIPT
position, the enlargement gradually dissipates as gravity takes effect. The term “wattle sign” is derived from the enlargement of the male turkey’s wattle as it becomes engorged with blood and
RI PT
functions to attract a female.
Imaging has proven to be a very significant tool in diagnosing the masseteric IMH, particularly if a phlebolith can be identified. When recognized in the presence of the somewhat
SC
limited clinical signs associated with the IMH, the phlebolith plays a key role in pointing to a diagnosis. The varied presence of a phlebolith (3-50% of IMHs) has been reported as an
M AN U
incidental imaging finding (1, 4, 12-15). Vessels subjected to abrupt stretching and rises in pressure during physical activity, such as mastication and exercise, can suffer damage to their intima (16). As part of the healing process, a protective thrombus develops at the point of injury. Alternatively, blood flowing through the tortuous vessels of a vascular lesion, tends to stagnate
TE D
and thrombose. The phlebolith develops when dystrophic calcification occurs within the thrombus. The phlebolith usually has a concentric lamellated pattern, a core that can be lucent or opaque, and consists of a mixture of calcium carbonate and calcium phosphate salts (17, 18).
EP
Small phleboliths tend to be uniformly opaque (19).
AC C
Standard panoramic radiography, in an office setting, can be helpful in uncovering the existence of a IMH phlebolith. However, the CT is the diagnostic approach of choice for phlebolith diagnosis. The scan derives its diagnostic ability from the fact that it is exquisitely sensitive to minute amounts of calcium which it can locate in three dimensions. Contrast should be avoided because its presence in a small vessel mimics the appearance of a phlebolith. Regardless, the CT scan is not the best imaging technique to uncover the presence of an IMH
4
ACCEPTED MANUSCRIPT
because even with contrast the vascularity of the IMH often is not adequately enhanced and phleboliths may be absent. However, some linear or spotty enhancement, reflecting vascularity, may be observed (20). Nevertheless, the CT scan is not sufficient to reliably clinch the presence
RI PT
and extent of vascularity or to demonstrate delineation between the IMH and muscle (10). Because only minimal enhancement is noted, the CT scan cannot convincingly suggest a
SC
diagnosis of IMH without the presence of a phlebolith.
The MRI with gadolinium has proven to be the diagnostic imaging procedure of choice
M AN U
for the IMH. It serves to accurately clarify the existence and location of the IMH within the masseter muscle, the IMH’s relation to adjacent anatomic structures, and when indicated determines the surgical approach. The IMH is isointense, similar to muscle, on a T1-weighted view. However, it enhances clearly, and is hyperintense on a T2-weighted study due to the free
TE D
water content of the vascularity present in the IMH. Low signals in the IMH are also seen in the T2-weighted film, and represent fibrous septae amid the vascular elements (21-25).
EP
The authors report a patient with a masseteric IMH who, other than a mild facial asymmetry, was subjectively asymptomatic. This report illustrates and emphasizes the central
AC C
role that imaging plays in the diagnosis of a masseteric IMH.
CASE REPORT
A 45-year-old female, in excellent health, was referred to the Columbia University Salivary Gland Center (SGC) because of painless swelling in the left parotid/masseteric region (Fig. 1). The patient stated that she has been aware of the facial swelling since the age of 4
5
ACCEPTED MANUSCRIPT
years. No treatment had been sought in the past because there were no subjective symptoms. The patient developed concerns regarding its persistent presence and the associated cosmetic
RI PT
asymmetry.
A CT scan had been previously requested by the patient’s physician (Fig.2). The report indicated the presence of an enlargement of the left masseter muscle.
A calcific density,
SC
measuring .5 x .4cm, was seen in the anterior medial aspect of the left masseter muscle. Abnormal enhancement within the masseter was also observed and thought to represent
M AN U
“postinflammatory/postinfectious change”. Because of the patient’s ensuing apprehension, she decided to seek further consultation in the SGC.
Clinically, a moderate left facial swelling was noted. The patient stated that the swelling
TE D
did not fluctuate in size or cause pain when eating in all the years of its known existence. She did state that the swelling seemed to be larger in the morning upon awakening from sleep, but would subside as the day progressed (the wattle sign). The swelling seemed to irregularly follow It was mostly anterior and inferior to the
EP
the anatomic contour of the masseter muscle.
superficial lobe of the parotid gland. Clenching accentuated the facial asymmetry. Palpation
AC C
indicated the enlargement was soft, painless and not circumscribed. The overlying skin and subcutaneous tissues were movable and there was no surface discoloration.
No cervical
lymphadenopathy was present.
Intraorally, the mucosa was moist. External massage of the left parotid salivary gland produced a normal clear salivary volume from a normal appearing left parotid duct orifice. No swelling or discoloration of the oral buccal mucosa were evident. A panoramic radiograph 6
ACCEPTED MANUSCRIPT
confirmed the presence of the calcific body previously identified by the CT scan (Fig. 3). It was located superimposed upon the anterior aspect of the left mandibular ramus, at the level of the
RI PT
occlusal plane of the mandibular molars.
A MRI with contrast was ordered for further evaluation and diagnosis (Fig. 4). The T2weighted MRI study clearly enhanced a vascular lesion within the left masseter muscle, and
SC
confirmed the presence and location of the phlebolith. A confident diagnosis of an IMH was now indicated. With the patient’s concerns regarding the existence of a neoplasm allayed, definitive
M AN U
therapeutic care was emphatically declined. She did state that she would be willing to return periodically for observation.
DISCUSSION
TE D
The means of diagnosing a masseteric IMH can not be achieved with any certainty from its location, duration, and presence of an asymptomatic swelling. In our patient, a convincing diagnosis could not be reached without the imaging studies. The CT scan identified the presence
EP
of enlargement and enhancement of the masseter, but the origin could not be determined. Clues were obtained from the presence of the calcified body noted on the CT scan and confirmed by
AC C
the panoramic radiograph. When this information was combined the lesion’s history, clinical examination and the T2-weighted MRI finding of a significantly enhanced masseter muscle from contained vascular fluid, a diagnosis of a vascular IMH was made possible.
Phleboliths are very uncommon in the oral region, and their presence suggests a vascular lesion. They can occur in the absence of such lesions, but they are mostly associated with vascular abnormalities. Our patient did not demonstrate the characteristic lamellated feature of a 7
ACCEPTED MANUSCRIPT
phlebolith. However, lamellation may be absent, as in our patient, in smaller phleboliths (19). Phleboliths are not to be confused with sialoliths. Their close proximity to the parotid gland leads to misdiagnosis. Differentiation rests in the absence of parotid sialadenitis and obstructive
RI PT
duct symptomatology. In comparison to sialoliths, phleboliths are usually multiple, more randomly located and lamellated. Furthermore, the ovality of the phlebolith can serve to distinguish it from the sialolith which is frequently elliptically shaped by the contour of the
SC
salivary duct. The IMH phlebolith must also be differentiated from other head and neck calicifications. The calcified lymph node often represents the end product of a lymphadenitis.
M AN U
Calcified acne lesions, atherosclerotic calcifications, residual phleboliths from involuted childhood hemangiomas, tonsilloliths, cysticercosis, and angioliths seen in some syndromes all must be considered.
TE D
The MRI represents the best imaging modality for diagnosis of the IMH. A T2-weighted film will show the IMH to have an intense signal while the surrounding tissues demonstrate low signal intensity. The T2 hyperintensity is due to the free water present in stagnant blood. The
EP
MRI also avoids ionizing radiation and demonstrates excellent contrast between a vascular lesion and adjacent tissues. However, phleboliths are not easily recognized on the T2-weighted film
AC C
because of their very low signal intensity. They are best identified on plain radiography and/or the CT scan.
8
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ILLUSTRATIONS
Clinical view of left facial swelling
Fig. 2.
CT scan (with contrast) demonstrates enlarged left masseter muscle
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Fig. 1.
containing phlebolith (black arrow). Contrast dye highlights vascular
Panoramic radiograph reveals phlebolith superimposed upon anterior
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Fig. 3.
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tissue (white arrow)
aspect of left mandibular ramus (arrow).
MRI with contrast (T2-weighted). Note hyperintense area involving left masseter muscle caused by IMH vascularity (arrowheads). Phlebolith
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(hypointense nodule) present (arrow).
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Fig. 4.
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