165
Breast Disease 34 (2014) 165–167 DOI 10.3233/BD-140370 IOS Press
Case Report
Multiple parenchymal leiomyomas of the breast in a Sudanese female Awad Ali M. Alawad Department of Surgery, Faculty of Medicine, University of Medical Sciences and Technology, Khartoum, Sudan Tel.: +249 912802545; E-mail: [email protected]
Abstract. Leiomyomas are benign smooth muscle neoplasm. They can occur in any organ, but the most common forms occur in the uterus, small bowel and the esophagus. Leiomyomas of breast are rare benign non epithelial tumor. Only 21 cases of solitary parenchymal leiomyoma of the breast have been reported. This is the first case of multiple breast leiomyomas being reported in the literature. Here we report a case of a 38-year-old woman who presented to us with a painless right sided breast swelling. Histopathology revealed a growth pattern of interlacing fascicles of smooth-muscle cells consistent with leiomyoma of breast. To the best of our knowledge, this is the first case of multiple intraparenchymal leiomyomas of the breast reported in the literature. Keywords: Breast, leiomyoma
1. Introduction Leiomyomas of the breast are one of the rarest benign nonepithelial tumors [1]. Most of them are found in the subareolar region. Leiomyoma of the breast parenchyma in the absence of periareolar lesions is uncommon [2]. Diagnosing these lesions as benign is essential for proper treatment. The origin of the intraparenchymal leiomyomas remains unclear. Theories include proliferation of smooth muscle cells surrounding blood vessels, teratoid origin with the extreme overgrowth of myomatous elements, differentiation from multipotent mesenchymal cells in breast tissue, and derivation from myoepithelial cells of breast ducts with clear differentiation to smooth muscle, or embryologically displaced smooth muscle cells from the nipple [3]. To the best of our knowledge, this is the first case of multiple intraparenchymal leiomyomas of the breast reported in the literature.
2. Case report A 38 –year- old lady complaining of right breast swelling was seen at the general surgery clinic of Uni-
Fig. 1. Large swelling of the right breast. (Colours are visible in the online version of the article; http://dx.doi.org/10.3233/BD-140370)
versity Charity Teaching Hospital. The swelling appeared two years ago and grew slowly with no pain or fever. There was no history of nipple discharge and she didn’t have any breast trauma. She took combined oral contraceptive drugs for 3 years. She didn’t lose weight over that period. She had no family history of breast cancer. She had her menarche at 12 yr of age and her first full term pregnancy at 22 yr of age. Physical examination revealed four mobile and well defined masses (largest one about 10 × 12 cm) involving the whole right breast (Fig. 1). They were firm and non tender with smooth surface. Axillary or supraclavicular lymph nodes were not palpable.
c 2011/2012 – IOS Press and the authors. All rights reserved 0888-6008/14/$27.50
166
A.A.M. Alawad / Multiple parenchymal leiomyomas of the breast in a Sudanese female
Fig. 2. Gross examination of surgical specimen revealed well encapsulated masses free from underlying breast tissue and overlying skin. (Colours are visible in the online version of the article; http://dx.doi. org/10.3233/BD-140370)
Mammography showed four oval masses with indistinct margins in the right breast. The masses were assessed as a BIRADS category 3. The masses were not visualized on ultrasonography. Provisional diagnosis of multiple adenomas was made. Fine needle aspiration cytology revealed benign mesenchymal tumor. Excision of the masses was done. Gross examination of the surgical specimen revealed well encapsulated masses free from underlying breast tissue and overlying skin (Fig. 2). Histopathological examination of the four masses revealed a growth pattern of interlacing fascicles of smooth-muscle cells consistent with leiomyoma of breast (Fig. 3). Post operative period was uneventful.
3. Discussion Leiomyomas are extremely rare breast tumors. Strong published the first description of this tumor in the breast parenchyma in 1913 [4]. Various theories have been proposed about the origin of these tumors. Kaufman suggested that they arise from the smoothmuscle cells that surround capillaries in the subcutaneous tissues of the breast [5]. Most breast leiomyomas are diagnosed in of middleaged women, and they usually occur in the right breast [6]. Our patient was in this age category and the leiomyomas developed in the parenchyma of her right breast. Most reported cases of solitary parenchymal leiomyoma gave no evidence of systemic disease or lymphadenopathy, but some complained of discomfort or pain. Differentiating benign intraparenchymal leiomyoma from other breast lesions is essential to determining the proper treatment. Tumors included in the pathologic differential diagnosis include leiomyosarcoma,
Fig. 3. Interlacing bundles of spindle shaped smooth muscle cells consistent with breast leiomyoma (HE X50). (Colours are visible in the online version of the article; http://dx.doi.org/10.3233/ BD-140370)
the spindle cell variant of adenomyoepithelioma, myofibroblastoma, fibromatosis, benign nerve sheath tumors including neurofibromas, and benign and malignant phyllodes tumors. Leiomyosarcoma is the most important lesion to be distinguished from an intraparenchymal leiomyoma as the treatment, recurrence rates, and prognosis differ. Since leiomyosarcoma and intraparenchymal leiomyomas share similar clinical and radiologic presentations, histologic examination is essential to definitively diagnose them. The common histopathologic features of leiomyomas of the breast are identical to those observed in leiomyomas at other sites: groups of interlacing bundles of spindleshaped cells with blunt-ended nuclei and eosinophilic cytoplasm [7]. In our patient, histopathologic findings indicated the masses were leiomyomas. The treatment for intraparenchymal leiomyoma of the breast is simple excision [8]. Due to the benign nature of these lesions, more extensive surgery is not indicated; however, as noted above, thorough histological examination is essential for a proper diagnosis. In previous studies, the possible link between tamoxifen, anti-obesity drugs and the growth of breast leiomyomas is questioned [9–11]. Our patient took oral contraceptive drugs for 3 years. Further studies are needed to confirm if there is any association. In conclusion, multiple intraparenchymal leiomyomas of the breast is an extremely rare disease. Clinical, radiological and Histopathological examinations are essential for diagnosis.
Conflict of interest None declared.
A.A.M. Alawad / Multiple parenchymal leiomyomas of the breast in a Sudanese female
References [7] [1]
[2]
[3]
[4] [5] [6]
Shah SD, Gupta A, Roy S, Mukhopadhyay S. Intraparenchymal leiomyoma of the breast: a case report. Indian J Surg. 2013 Jun; 75(Suppl 1): 88-9. Minami S, Matsuo S, Azuma T, Uga T, Hayashi T, Eguchi S, et al. Parenchymal leiomyoma of the breast: A case report with special reference to magnetic resonance imaging findings and an update review of literature. Breast Cancer. 2011 Jul; 18(3): 231-6. Koirala K, Shrestha ML, Chalise PR, Shrestha BB, Shrestha R. Leiomyoma of breast: a report of rare case. Nepal Med Coll J. 2008 Sep; 10(3): 207-8. Weldon C, Jones B, Daroca P, Beech D. Breast leiomyoma. J La State Med Soc. 1998 Aug; 150(8): 367-70. Kaufman HL, Hirsch EF. Leiomyoma of the breast. J Surg Oncol. 1996 May; 62(1): 62-4. Pourbagher A, Pourbagher MA, Bal N, Oguzkurt L, Ezer A. Leiomyoma of the breast parenchyma. AJR Am J Roentgenol.
[8]
[9] [10]
[11]
167
2005 Dec; 185(6): 1595-7. Ende L, Mercado C, Axelrod D, Darvishian F, Levine P, Cangiarella J. Intraparenchymal leiomyoma of the breast: A case report and review of the literature. Ann Clin Lab Sci. 2007 Summer; 37(3): 268-73. Heyer H, Ohlinger R, Schimming A, Schwesinger G, Grunwald S. Parenchymal leiomyoma of the breast–clinical, sonographic, mammographic and histological features. Ultraschall Med. 2006 Feb; 27(1): 55-8. Abd el-All HS. Breast spindle cell tumours: About eight cases. Diagn Pathol. 2006; 1: 13. Kotsuma Y, Wakasa K, Yayoi E, Kishibuchi M, Sakamoto G. A case of leiomyoma of the breast. Breast Cancer. 2001; 8(2): 166-9. Vecchio GM, Cavaliere A, Cartaginese F, Lucaccioni A, Lombardi T, Parenti R, et al. Intraparenchymal leiomyoma of the breast: report of a case with emphasis on needle core biopsybased diagnosis. Pathologica. 2013 Aug; 105(4): 122-7.
Copyright of Breast Disease is the property of IOS Press and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Breast Disease 34 (2014) 165–167 DOI 10.3233/BD-140370 IOS Press
Case Report
Multiple parenchymal leiomyomas of the breast in a Sudanese female Awad Ali M. Alawad Department of Surgery, Faculty of Medicine, University of Medical Sciences and Technology, Khartoum, Sudan Tel.: +249 912802545; E-mail: [email protected]
Abstract. Leiomyomas are benign smooth muscle neoplasm. They can occur in any organ, but the most common forms occur in the uterus, small bowel and the esophagus. Leiomyomas of breast are rare benign non epithelial tumor. Only 21 cases of solitary parenchymal leiomyoma of the breast have been reported. This is the first case of multiple breast leiomyomas being reported in the literature. Here we report a case of a 38-year-old woman who presented to us with a painless right sided breast swelling. Histopathology revealed a growth pattern of interlacing fascicles of smooth-muscle cells consistent with leiomyoma of breast. To the best of our knowledge, this is the first case of multiple intraparenchymal leiomyomas of the breast reported in the literature. Keywords: Breast, leiomyoma
1. Introduction Leiomyomas of the breast are one of the rarest benign nonepithelial tumors [1]. Most of them are found in the subareolar region. Leiomyoma of the breast parenchyma in the absence of periareolar lesions is uncommon [2]. Diagnosing these lesions as benign is essential for proper treatment. The origin of the intraparenchymal leiomyomas remains unclear. Theories include proliferation of smooth muscle cells surrounding blood vessels, teratoid origin with the extreme overgrowth of myomatous elements, differentiation from multipotent mesenchymal cells in breast tissue, and derivation from myoepithelial cells of breast ducts with clear differentiation to smooth muscle, or embryologically displaced smooth muscle cells from the nipple [3]. To the best of our knowledge, this is the first case of multiple intraparenchymal leiomyomas of the breast reported in the literature.
2. Case report A 38 –year- old lady complaining of right breast swelling was seen at the general surgery clinic of Uni-
Fig. 1. Large swelling of the right breast. (Colours are visible in the online version of the article; http://dx.doi.org/10.3233/BD-140370)
versity Charity Teaching Hospital. The swelling appeared two years ago and grew slowly with no pain or fever. There was no history of nipple discharge and she didn’t have any breast trauma. She took combined oral contraceptive drugs for 3 years. She didn’t lose weight over that period. She had no family history of breast cancer. She had her menarche at 12 yr of age and her first full term pregnancy at 22 yr of age. Physical examination revealed four mobile and well defined masses (largest one about 10 × 12 cm) involving the whole right breast (Fig. 1). They were firm and non tender with smooth surface. Axillary or supraclavicular lymph nodes were not palpable.
c 2011/2012 – IOS Press and the authors. All rights reserved 0888-6008/14/$27.50
166
A.A.M. Alawad / Multiple parenchymal leiomyomas of the breast in a Sudanese female
Fig. 2. Gross examination of surgical specimen revealed well encapsulated masses free from underlying breast tissue and overlying skin. (Colours are visible in the online version of the article; http://dx.doi. org/10.3233/BD-140370)
Mammography showed four oval masses with indistinct margins in the right breast. The masses were assessed as a BIRADS category 3. The masses were not visualized on ultrasonography. Provisional diagnosis of multiple adenomas was made. Fine needle aspiration cytology revealed benign mesenchymal tumor. Excision of the masses was done. Gross examination of the surgical specimen revealed well encapsulated masses free from underlying breast tissue and overlying skin (Fig. 2). Histopathological examination of the four masses revealed a growth pattern of interlacing fascicles of smooth-muscle cells consistent with leiomyoma of breast (Fig. 3). Post operative period was uneventful.
3. Discussion Leiomyomas are extremely rare breast tumors. Strong published the first description of this tumor in the breast parenchyma in 1913 [4]. Various theories have been proposed about the origin of these tumors. Kaufman suggested that they arise from the smoothmuscle cells that surround capillaries in the subcutaneous tissues of the breast [5]. Most breast leiomyomas are diagnosed in of middleaged women, and they usually occur in the right breast [6]. Our patient was in this age category and the leiomyomas developed in the parenchyma of her right breast. Most reported cases of solitary parenchymal leiomyoma gave no evidence of systemic disease or lymphadenopathy, but some complained of discomfort or pain. Differentiating benign intraparenchymal leiomyoma from other breast lesions is essential to determining the proper treatment. Tumors included in the pathologic differential diagnosis include leiomyosarcoma,
Fig. 3. Interlacing bundles of spindle shaped smooth muscle cells consistent with breast leiomyoma (HE X50). (Colours are visible in the online version of the article; http://dx.doi.org/10.3233/ BD-140370)
the spindle cell variant of adenomyoepithelioma, myofibroblastoma, fibromatosis, benign nerve sheath tumors including neurofibromas, and benign and malignant phyllodes tumors. Leiomyosarcoma is the most important lesion to be distinguished from an intraparenchymal leiomyoma as the treatment, recurrence rates, and prognosis differ. Since leiomyosarcoma and intraparenchymal leiomyomas share similar clinical and radiologic presentations, histologic examination is essential to definitively diagnose them. The common histopathologic features of leiomyomas of the breast are identical to those observed in leiomyomas at other sites: groups of interlacing bundles of spindleshaped cells with blunt-ended nuclei and eosinophilic cytoplasm [7]. In our patient, histopathologic findings indicated the masses were leiomyomas. The treatment for intraparenchymal leiomyoma of the breast is simple excision [8]. Due to the benign nature of these lesions, more extensive surgery is not indicated; however, as noted above, thorough histological examination is essential for a proper diagnosis. In previous studies, the possible link between tamoxifen, anti-obesity drugs and the growth of breast leiomyomas is questioned [9–11]. Our patient took oral contraceptive drugs for 3 years. Further studies are needed to confirm if there is any association. In conclusion, multiple intraparenchymal leiomyomas of the breast is an extremely rare disease. Clinical, radiological and Histopathological examinations are essential for diagnosis.
Conflict of interest None declared.
A.A.M. Alawad / Multiple parenchymal leiomyomas of the breast in a Sudanese female
References [7] [1]
[2]
[3]
[4] [5] [6]
Shah SD, Gupta A, Roy S, Mukhopadhyay S. Intraparenchymal leiomyoma of the breast: a case report. Indian J Surg. 2013 Jun; 75(Suppl 1): 88-9. Minami S, Matsuo S, Azuma T, Uga T, Hayashi T, Eguchi S, et al. Parenchymal leiomyoma of the breast: A case report with special reference to magnetic resonance imaging findings and an update review of literature. Breast Cancer. 2011 Jul; 18(3): 231-6. Koirala K, Shrestha ML, Chalise PR, Shrestha BB, Shrestha R. Leiomyoma of breast: a report of rare case. Nepal Med Coll J. 2008 Sep; 10(3): 207-8. Weldon C, Jones B, Daroca P, Beech D. Breast leiomyoma. J La State Med Soc. 1998 Aug; 150(8): 367-70. Kaufman HL, Hirsch EF. Leiomyoma of the breast. J Surg Oncol. 1996 May; 62(1): 62-4. Pourbagher A, Pourbagher MA, Bal N, Oguzkurt L, Ezer A. Leiomyoma of the breast parenchyma. AJR Am J Roentgenol.
[8]
[9] [10]
[11]
167
2005 Dec; 185(6): 1595-7. Ende L, Mercado C, Axelrod D, Darvishian F, Levine P, Cangiarella J. Intraparenchymal leiomyoma of the breast: A case report and review of the literature. Ann Clin Lab Sci. 2007 Summer; 37(3): 268-73. Heyer H, Ohlinger R, Schimming A, Schwesinger G, Grunwald S. Parenchymal leiomyoma of the breast–clinical, sonographic, mammographic and histological features. Ultraschall Med. 2006 Feb; 27(1): 55-8. Abd el-All HS. Breast spindle cell tumours: About eight cases. Diagn Pathol. 2006; 1: 13. Kotsuma Y, Wakasa K, Yayoi E, Kishibuchi M, Sakamoto G. A case of leiomyoma of the breast. Breast Cancer. 2001; 8(2): 166-9. Vecchio GM, Cavaliere A, Cartaginese F, Lucaccioni A, Lombardi T, Parenti R, et al. Intraparenchymal leiomyoma of the breast: report of a case with emphasis on needle core biopsybased diagnosis. Pathologica. 2013 Aug; 105(4): 122-7.
Copyright of Breast Disease is the property of IOS Press and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
