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Intra-articular angioleiomyoma of the knee: A case report
The Knee 13 (2006) 330 – 332 www.elsevier.com/locate/knee
Short communication
Intra-articular angioleiomyoma of the knee: A case reportB Kohjirou Okahashi a,*, Kazuya Sugimoto a,b, Makoto Iwai a,b, Manabu Oshima a,b, Yoshinori Takakura a,c a
Medical Center for Emergency and Critical Care, Nara Prefectural Nara Hospital 1-30-1, Hiramatsu-cho, Nara-city, Nara 631-0846, Japan b Department of Orthopaedic Surgery, Saiseikai Nara Hospital, 4-643 Hachijo, Nara-shi, Nara 6308145, Japan c Department of Orthopaedic Surgery, Nara Medical University, Nara, Japan Received 8 December 2005; received in revised form 28 February 2006; accepted 8 March 2006
Abstract We report a rare case of angioleiomyoma that occurred in the intra-articular portion of the knee joint. A 43-year-old female was referred to us with a 3-year history of recurrent pain and a loss of full extension of the right knee. Physical examinations revealed swelling and restriction of active full extension. Magnetic resonance imaging (MRI) revealed an intra-articular tumor. Surgical excision was performed, and the histology was characteristic of an angioleiomyoma. The patient became asymptomatic after the operation. At 2-year follow-up after the operation, no recurrence has developed. D 2006 Elsevier B.V. All rights reserved. Keywords: Angioleiomyoma; Intra-articular; Knee; Magnetic resonance imaging (MRI)
1. Introduction Angioleiomyomas are benign soft tissue tumors with a predilection for the lower extremities in middle-aged females [4,9]. But angioleiomyoma of the knee joint is very rare [7]. Atypical locations of the tumor add to the difficulty of making a diagnosis. To avoid making a misdiagnosis, a high index of suspicion is required to make the diagnosis. Misdiagnosis delays appropriate treatment. Angioleiomyomas should be included in the differential diagnosis of intra-articular tumors.
2. Clinical history A 43-year-old female was referred to our hospital with a 3year history of recurrent pain and extension loss in the right knee under the diagnosis of meniscus injury. There was minor i
Investigation performed at the Department of Orthopaedic Surgery, Saiseikai Nara Hospital, Nara, Japan. * Corresponding author. Tel.: +81 742 46 6060; fax: +81 742 46 6510. E-mail address: [email protected] (K. Okahashi). 0968-0160/$ - see front matter D 2006 Elsevier B.V. All rights reserved. doi:10.1016/j.knee.2006.03.002
history of trauma. She had felt knee pain at landing from a jump 3years before. The symptom was mitigated by local rest, and she did not consult any physician. But she could not achieve full extension gradually in the right knee. She consulted a local physician, who noted restriction of passive extension and hemarthrosis. The symptoms were not mitigated by any conservative treatment including NASIDS medication, injection of corticosteroid, application of orthosis and physical therapies. Physical examination of the affected knee revealed a loss of passive full extension ( 15-) and swelling in the whole right knee. There was slight muscle atrophy in the affected thigh, but typical signs of meniscus lesion were not detected. Routine radiography showed no remarkable changes. Laboratory data were normal. MRI revealed an intra-articular mass of the knee joint, measuring 5 cm in diameter. The intensity of the mass was homogenous with isointense to muscle on T1-weighted sequences and heterogeneous intensity specks are noted within the mass on T2-weighted sequences (Fig. 1A,B,C). Arthroscopic examination was performed to confirm the nature, location and extent of the mass, but grasp of a total image was impossible.
K. Okahashi et al. / The Knee 13 (2006) 330 – 332
331
Fig. 2. Clinical appearance of the tumor.
adhesive partially to the anterior joint capsule. Complete excision of the mass was performed. The excised mass was a macroscopically lustrous, smooth-surfaced, elastic soft, white and red colored mass, measuring 5 4 2 cm (Fig. 3). Microscopic findings revealed that the tumor was composed of numerous blood vessels of various sizes and copious fascicles of smooth muscle bundle surrounding the vessels. The histology was characteristic of angioleiomyoma (Fig. 4). The patient became asymptomatic immediately after the operation. Recurrence of the tumor has not been detected as of 2 years after the operation.
3. Discussion Angioleiomyomas are benign solitary tumors of smooth muscle origin that arise from the muscular layer of vessel walls. Since Stout published the first comprehensive review of this rare lesion in 1937 [11], although predominantly found in the lower extremities, several angioleiomyomas around the knee have also been reported [2,6,12]. But few cases in which an angioleiomyoma occurred within an intra-
Fig. 1. MR images demonstrate an intra-articular mass in the knee joint. T1weighted MRI reveals the mass is homogenous with isointense to muscle. Heterogeneous signal intensity specks are noted within the mass on T2weighted MRI. (A) Sagittal T1-weighted MRI. (B) Axial T1-weighted MRI. (C) Axial T2-weighted MRI.
A 5-cm-long skin incision was made just lateral to the patellar tendon. The mass was found mainly in the intercondylar notch of the knee joint and protruded to the lateral joint side (Fig. 2). The mass was capsulized and
Fig. 3. The mass after resection, measuring 5 4 2cm.
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K. Okahashi et al. / The Knee 13 (2006) 330 – 332
Fig. 4. Microscopic specimen demonstrates angioleiomyoma. Microscopic findings revealed that the tumor was composed of numerous fascicles of smooth muscle bundles and blood vessels (haematoxylin and eosin, 10).
investigations such as arthroscopy are recommended to confirm the histological character of the lesion. However, MRI in patients with any suspected intraarticular tumors may decrease the time until diagnosis and MRI should be used as this is a noninvasive and useful technique which can provide much preoperative information [7,9]. All angioleiomyomas are benign and are adequately treated by simple excision. Our patient became completely free of her preoperative symptoms, and no recurrence had occurred at 2-year follow-up. If a middle-aged female complains of nonspecific symptoms such as intermittent pain and hemarthrosis in the affected knee, we should be aware of atypical locations of angioleiomyoma to avoid misdiagnosis.
Acknowledgement articular portion of the knee joint have been reported [7]. Atypical locations of the tumor add to the difficulty of making a diagnosis. In most of these reports, much time elapsed before an intra-articular tumor could be correctly diagnosed [7,9]. Usually an angioleiomyoma presents as a painful, solitary, subcutaneous lesion located in the soft palate, eyelid, external auditory canal, urethra and hands or feet [4,8]. In the current case, the tumor was located in the intraarticular portion of the knee joint, therefore much time elapsed before the patient developed subjective symptoms. Intra-articular tumors are very unusual and physicians are apt to misdiagnose conditions like meniscus tear or arthritis [9]. It took 3years from the first visit to a local doctor until the final and correct diagnosis of the lesion was made. To avoid making a misdiagnosis a high index of suspicion is required. For the differential diagnosis, all intra-articular tumors such as pigmented villonodular synovitis (PVS), hemangioma, synovial chondromatosis, lipoma, and ganglion have to be considered, particularly PVS. PVS has been subclassified into diffuse or nodular forms based on the macroscopic appearance [5]. Local excision of nodular PVS usually results in complete relief of symptoms without recurrence [1,5]. Curing patients with diffuse disease is more difficult, with recurrence rates of up to 46% after complete synovectomy, as reported by Byers et al. [1]. It is difficult to give a correct diagnosis based on the imaging findings. The MRI findings and clinical symptoms may be similar in all intra-articular tumors. Caution must be taken, because MRI is useful but not specific in the diagnosis of PVS [3,10]. Since preoperative verification of the histological diagnosis is impossible, early invasive
No benefits in any form have been received or will be received from any commercial party related directly of indirectly to the subject of this article. No funds were received in support of this study.
References [1] Byers PD, Cotton RE, Deacon OW, Lowy M, Newman PH, Sissons HA, et al. The diagnosis and treatment of pigmented villonodular synovitis. J Bone Joint Surg, Am Br Vol 1968;50:290 – 305. [2] Dicaprio MR, Jokl P. Vascular leiomyoma presenting as medial joint line pain of the knee. Arthroscopy 2003;19:1 – 3. [3] Durr HR, Stabler A, Maier M, Refior HJ. Pigmented villonodular synovitis. Review of 20 cases. J Rheumatol 2001;28:1620 – 30. [4] Enzinger FM, Weiss SW. Benign tumors of smooth muscle & Glomus Tumors. Soft Tissue Tumors. 2nd ed. St. Louis: CV Mosby Co; 1988 383 – 401 and 581 – 595. [5] Granowitz SP, D’Antonio J, Mankin HL. The pathogenesis and longterm end results of pigmented villonodular synovitis. Clin Orthop Relat Res 1976;114:335 – 51. [6] Gassel F, Sommer T, Meybehm M, Schmitt O. A case of unusual space-occupying lesion in the knee joint. Radiologe 1999;39:703 – 5. [7] Gulati MS, Kapoor A, Maheshwari J. Angiomyoma of the knee joint: value of magnetic resonance imaging. Australas Radiol 1999;43: 353 – 4. [8] Kinoshita T, Ishii K, Abe Y, Naganuma H. Angiomyoma of the lower extremity: MR findings. Skelet Radiol 1997;26:443 – 5. [9] Okahashi K, Sugimoto K, Iwai M, Tanaka M, Fujisawa Y, Takakura Y. Intra-articular synovial hemangioma; a rare cause of knee pain and swelling. Arch Orthop Trauma Surg 2004;124:571 – 3. [10] Poletti SC, Gates III HS, Martinez SM, Richardson WJ. The use of magnetic resonance imaging in the diagnosis of pigmented villonodular synovitis. Orthopedics 1990;13:185 – 90. [11] Stout AP. Solitary cutaneous and subcutaneous leiomyoma. Am J Cancer 1937;29:435 – 69. [12] Thienpont E, Geens S, Nelen G. Angioleiomyoma of the knee. A case report. Acta Orthop Belg 2002;68:76 – 8.
Short communication
Intra-articular angioleiomyoma of the knee: A case reportB Kohjirou Okahashi a,*, Kazuya Sugimoto a,b, Makoto Iwai a,b, Manabu Oshima a,b, Yoshinori Takakura a,c a
Medical Center for Emergency and Critical Care, Nara Prefectural Nara Hospital 1-30-1, Hiramatsu-cho, Nara-city, Nara 631-0846, Japan b Department of Orthopaedic Surgery, Saiseikai Nara Hospital, 4-643 Hachijo, Nara-shi, Nara 6308145, Japan c Department of Orthopaedic Surgery, Nara Medical University, Nara, Japan Received 8 December 2005; received in revised form 28 February 2006; accepted 8 March 2006
Abstract We report a rare case of angioleiomyoma that occurred in the intra-articular portion of the knee joint. A 43-year-old female was referred to us with a 3-year history of recurrent pain and a loss of full extension of the right knee. Physical examinations revealed swelling and restriction of active full extension. Magnetic resonance imaging (MRI) revealed an intra-articular tumor. Surgical excision was performed, and the histology was characteristic of an angioleiomyoma. The patient became asymptomatic after the operation. At 2-year follow-up after the operation, no recurrence has developed. D 2006 Elsevier B.V. All rights reserved. Keywords: Angioleiomyoma; Intra-articular; Knee; Magnetic resonance imaging (MRI)
1. Introduction Angioleiomyomas are benign soft tissue tumors with a predilection for the lower extremities in middle-aged females [4,9]. But angioleiomyoma of the knee joint is very rare [7]. Atypical locations of the tumor add to the difficulty of making a diagnosis. To avoid making a misdiagnosis, a high index of suspicion is required to make the diagnosis. Misdiagnosis delays appropriate treatment. Angioleiomyomas should be included in the differential diagnosis of intra-articular tumors.
2. Clinical history A 43-year-old female was referred to our hospital with a 3year history of recurrent pain and extension loss in the right knee under the diagnosis of meniscus injury. There was minor i
Investigation performed at the Department of Orthopaedic Surgery, Saiseikai Nara Hospital, Nara, Japan. * Corresponding author. Tel.: +81 742 46 6060; fax: +81 742 46 6510. E-mail address: [email protected] (K. Okahashi). 0968-0160/$ - see front matter D 2006 Elsevier B.V. All rights reserved. doi:10.1016/j.knee.2006.03.002
history of trauma. She had felt knee pain at landing from a jump 3years before. The symptom was mitigated by local rest, and she did not consult any physician. But she could not achieve full extension gradually in the right knee. She consulted a local physician, who noted restriction of passive extension and hemarthrosis. The symptoms were not mitigated by any conservative treatment including NASIDS medication, injection of corticosteroid, application of orthosis and physical therapies. Physical examination of the affected knee revealed a loss of passive full extension ( 15-) and swelling in the whole right knee. There was slight muscle atrophy in the affected thigh, but typical signs of meniscus lesion were not detected. Routine radiography showed no remarkable changes. Laboratory data were normal. MRI revealed an intra-articular mass of the knee joint, measuring 5 cm in diameter. The intensity of the mass was homogenous with isointense to muscle on T1-weighted sequences and heterogeneous intensity specks are noted within the mass on T2-weighted sequences (Fig. 1A,B,C). Arthroscopic examination was performed to confirm the nature, location and extent of the mass, but grasp of a total image was impossible.
K. Okahashi et al. / The Knee 13 (2006) 330 – 332
331
Fig. 2. Clinical appearance of the tumor.
adhesive partially to the anterior joint capsule. Complete excision of the mass was performed. The excised mass was a macroscopically lustrous, smooth-surfaced, elastic soft, white and red colored mass, measuring 5 4 2 cm (Fig. 3). Microscopic findings revealed that the tumor was composed of numerous blood vessels of various sizes and copious fascicles of smooth muscle bundle surrounding the vessels. The histology was characteristic of angioleiomyoma (Fig. 4). The patient became asymptomatic immediately after the operation. Recurrence of the tumor has not been detected as of 2 years after the operation.
3. Discussion Angioleiomyomas are benign solitary tumors of smooth muscle origin that arise from the muscular layer of vessel walls. Since Stout published the first comprehensive review of this rare lesion in 1937 [11], although predominantly found in the lower extremities, several angioleiomyomas around the knee have also been reported [2,6,12]. But few cases in which an angioleiomyoma occurred within an intra-
Fig. 1. MR images demonstrate an intra-articular mass in the knee joint. T1weighted MRI reveals the mass is homogenous with isointense to muscle. Heterogeneous signal intensity specks are noted within the mass on T2weighted MRI. (A) Sagittal T1-weighted MRI. (B) Axial T1-weighted MRI. (C) Axial T2-weighted MRI.
A 5-cm-long skin incision was made just lateral to the patellar tendon. The mass was found mainly in the intercondylar notch of the knee joint and protruded to the lateral joint side (Fig. 2). The mass was capsulized and
Fig. 3. The mass after resection, measuring 5 4 2cm.
332
K. Okahashi et al. / The Knee 13 (2006) 330 – 332
Fig. 4. Microscopic specimen demonstrates angioleiomyoma. Microscopic findings revealed that the tumor was composed of numerous fascicles of smooth muscle bundles and blood vessels (haematoxylin and eosin, 10).
investigations such as arthroscopy are recommended to confirm the histological character of the lesion. However, MRI in patients with any suspected intraarticular tumors may decrease the time until diagnosis and MRI should be used as this is a noninvasive and useful technique which can provide much preoperative information [7,9]. All angioleiomyomas are benign and are adequately treated by simple excision. Our patient became completely free of her preoperative symptoms, and no recurrence had occurred at 2-year follow-up. If a middle-aged female complains of nonspecific symptoms such as intermittent pain and hemarthrosis in the affected knee, we should be aware of atypical locations of angioleiomyoma to avoid misdiagnosis.
Acknowledgement articular portion of the knee joint have been reported [7]. Atypical locations of the tumor add to the difficulty of making a diagnosis. In most of these reports, much time elapsed before an intra-articular tumor could be correctly diagnosed [7,9]. Usually an angioleiomyoma presents as a painful, solitary, subcutaneous lesion located in the soft palate, eyelid, external auditory canal, urethra and hands or feet [4,8]. In the current case, the tumor was located in the intraarticular portion of the knee joint, therefore much time elapsed before the patient developed subjective symptoms. Intra-articular tumors are very unusual and physicians are apt to misdiagnose conditions like meniscus tear or arthritis [9]. It took 3years from the first visit to a local doctor until the final and correct diagnosis of the lesion was made. To avoid making a misdiagnosis a high index of suspicion is required. For the differential diagnosis, all intra-articular tumors such as pigmented villonodular synovitis (PVS), hemangioma, synovial chondromatosis, lipoma, and ganglion have to be considered, particularly PVS. PVS has been subclassified into diffuse or nodular forms based on the macroscopic appearance [5]. Local excision of nodular PVS usually results in complete relief of symptoms without recurrence [1,5]. Curing patients with diffuse disease is more difficult, with recurrence rates of up to 46% after complete synovectomy, as reported by Byers et al. [1]. It is difficult to give a correct diagnosis based on the imaging findings. The MRI findings and clinical symptoms may be similar in all intra-articular tumors. Caution must be taken, because MRI is useful but not specific in the diagnosis of PVS [3,10]. Since preoperative verification of the histological diagnosis is impossible, early invasive
No benefits in any form have been received or will be received from any commercial party related directly of indirectly to the subject of this article. No funds were received in support of this study.
References [1] Byers PD, Cotton RE, Deacon OW, Lowy M, Newman PH, Sissons HA, et al. The diagnosis and treatment of pigmented villonodular synovitis. J Bone Joint Surg, Am Br Vol 1968;50:290 – 305. [2] Dicaprio MR, Jokl P. Vascular leiomyoma presenting as medial joint line pain of the knee. Arthroscopy 2003;19:1 – 3. [3] Durr HR, Stabler A, Maier M, Refior HJ. Pigmented villonodular synovitis. Review of 20 cases. J Rheumatol 2001;28:1620 – 30. [4] Enzinger FM, Weiss SW. Benign tumors of smooth muscle & Glomus Tumors. Soft Tissue Tumors. 2nd ed. St. Louis: CV Mosby Co; 1988 383 – 401 and 581 – 595. [5] Granowitz SP, D’Antonio J, Mankin HL. The pathogenesis and longterm end results of pigmented villonodular synovitis. Clin Orthop Relat Res 1976;114:335 – 51. [6] Gassel F, Sommer T, Meybehm M, Schmitt O. A case of unusual space-occupying lesion in the knee joint. Radiologe 1999;39:703 – 5. [7] Gulati MS, Kapoor A, Maheshwari J. Angiomyoma of the knee joint: value of magnetic resonance imaging. Australas Radiol 1999;43: 353 – 4. [8] Kinoshita T, Ishii K, Abe Y, Naganuma H. Angiomyoma of the lower extremity: MR findings. Skelet Radiol 1997;26:443 – 5. [9] Okahashi K, Sugimoto K, Iwai M, Tanaka M, Fujisawa Y, Takakura Y. Intra-articular synovial hemangioma; a rare cause of knee pain and swelling. Arch Orthop Trauma Surg 2004;124:571 – 3. [10] Poletti SC, Gates III HS, Martinez SM, Richardson WJ. The use of magnetic resonance imaging in the diagnosis of pigmented villonodular synovitis. Orthopedics 1990;13:185 – 90. [11] Stout AP. Solitary cutaneous and subcutaneous leiomyoma. Am J Cancer 1937;29:435 – 69. [12] Thienpont E, Geens S, Nelen G. Angioleiomyoma of the knee. A case report. Acta Orthop Belg 2002;68:76 – 8.