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Mammary duct ectasia in children presenting bloody nipple discharge: A case in a pubertal girl
Mammary Duct Ectasia in Children Presenting Bloody Nipple Discharge: A Case in a Pubertal Girl By Shinzo Kitahara, Mineo Wakabayashi, Tadaaki Shiba, Kyoei Nonaka, Hiroko Nonaka, and Izumi Kobayashi Tokyo, Japan
Mammary duct ectasia occurs rarely in childhood. The authors report on the case of a pubertal girl who was operated on for duct ectasia with bloody nipple discharge. Duct ectasia is regarded as a primary lesion; it is considered to be a cause of bloody secretion, and it has a mechanism similar to that of mammary duct papilloma.
J Pediatr Surg 36:E2. Copyright Company.
M
of the above-mentioned evidence, the girl was diagnosed as having a possible case of mammary duct ectasia.
AMMARY DUCT ECTASIA is one of the causes of abnormal nipple discharge, usually found in perimenopausal para, and it rarely occurs in childhood. Its cause is unknown. We observed a case in a 10-yearold girl of mammary duct ectasia. The patient came to the hospital complaining chiefly of bloody nipple discharge. Here we report our findings with a review of the literature. CASE REPORT On September 28, 1998, a premenarcheal girl, 10 years, 8 months old, visited our clinic complaining of bloody discharge from the left nipple. No problem was recognized in her medical history, family history, or development. In the cytology of the secretion, only red blood cells and foam cells were found, and epithelial cells were not recognized. In ultrasonography and computed tomography (CT) scan, marked dilation of the mammary ducts was observed, extending to the periphery from just beneath the nipple, but no clear tumor images were found (Fig 1). There were no abnormal findings in the blood or biochemical test results, blood hormone level, or thyroid function findings. In bacterial cultures of the secreta, Streptococcus spp. were detected on the first occasion and Staphylococcus epidermidis on the second. However, the possibility of bacterial contamination could not be ruled out, because there was no clinical sign of infection. Therefore, antibacterial therapy was not carried out. Because she was in her growth phase, and because no malignancy was observed with cytology, follow-up was carried out. She had her menarche during the follow-up. Because the nipple discharge continued, and no change was seen in to imaging, surgery was performed on July 23, 1999. Arc incision of approximately a semicircle was made in the lower half of the left areola, and extraction of the dilated ducts, along with a little of the breast tissue, was carried out. Microscopic findings (Fig 2) showed that the epithelium of the markedly dilated mammary duct had approximately 2 layers of columnar epithelium and myoepithelial cells. Slight papillary erythrocytes and degenerated necrotic tissue were observed inside the dilated mammary duct, with proliferation in several areas. There was a minor amount of small round cell infiltration in the subepithelium of the mammary duct. Strong hemostasis and fibrotic infiltration were noticed in several parts of the stroma. Hemosiderosis and a small amount of inflammation histiocyte collection were noticed in only one place, but mammary duct damage was not found in this area. In the mammary duct, no remarkable intraluminal obliteration was noticed. On the basis Journal of Pediatric Surgery, Vol 36, No 6 (June), 2001: E2
©
2001 by W.B. Saunders
INDEX WORDS: Mammary duct ectasia, bloody nipple discharge, puberty.
DISCUSSION
The term mammary duct ectasia was proposed by Haagensen (1951)1 for benign lesion of the breast consisting of dilation of the mammary duct, periductal fibrosis, and inflammation, and is accepted widely today. Mammary duct ectasia commonly occurs during perimenopausal multipara,2 but it occurs rarely in nullipara,3 men,4,5 and children.6,7 Regarding mammary duct ectasia of children, no reports of occurrences in pubertal girls are found, although there are reports of occurrences in boys6 and infants.7 In these cases, cause of duct ectasia could not be specified, but the lesion was unilateral. Hormone abnormality was not recognized, and the obstruction finding was not histologically seen in the collecting tubules in the vicinity of the nipple. There are some reports8,9 that bacterial infection is the cause. In this case, bacterial culture of the nipple secretion was positive, but clinical and histologic finding of abscess and other forms of inflammation were lacking. Although the possibility of contamination could not be ruled out, there did not seem to be an association with the cause of disease. Variability of histologic findings of mammary duct ectasia is regarded as depending on the stage of the disease,10 although there is controversy as to which condition is the first development of the inflammatory change of stroma and dilation of mammary duct.2 Dixon et al From the Second Department of Surgery, Department of Pathology, Faculty of Medicine, Toho University, and the Department of Surgery, Kobayashi Hospital, Tokyo, Japan. Address reprint requests to Shinzo Kitahara, Second Department of Surgery, Faculty of Medicine, Toho University, 6-11-1, Omori Nishi, Otaku, Tokyo 143-8541, Japan. Copyright © 2001 by W.B. Saunders Company 1531-5037/01/3606-0040$35.00/0 doi:10.1053/jpsu.2001.24011 1
2
KITAHARA ET AL
Fig 1. Ultrasonographic findings: Mammary ducts in radiating dilation are visualized around nipple.
Fig 2. Histologically, erythrocytes and necrotic tissue are found in the lumen of the mammary duct, which is dilated markedly, and hemosiderin leakage is noticeable in the stroma. The duct epithelium shows papillary growth in some areas.
(1986)10 suggested that periductal mastitis preceded mammary duct ectasia, and that it was the first change in this condition, based on the result that surrounding periductal inflammation of the nondilated duct is marked in younger patients. In older patients, however, marked duct ectasia accompanied by nipple retraction often is observed. However, according to findings to the effect that almost no inflammatory cell infiltration of stroma is found even if mammary duct dilation is markedly revealed and leakage of hemosiderin from mammary duct
to stroma is recognized, it is thought that duct ectasia is the primary lesion. Some areas with papillary growth are found in dilated mammary duct epithelium, and so the mechanism is considered to be a cause of bloody discharge, as in mammary duct papilloma. Considering that the girl was in her growth period, a less invasive method, involving little breast deformation was deemed appropriate. It was thought that a skin incision in the surrounding areola and extirpation of dilated ducts should be satisfactory in this respect.
REFERENCES 1. Haagensen CD: Mammary duct ectasia: A disease that may simulate carcinoma. Cancer 4:749-761, 1951 2. Millis RR: In Gresham GA (ed): Atlas of Breast Pathology vol 7. Lancaster, England, MTP Press Limited, 1984, pp 21-23 3. Rees BI, Gravelle, IH, Hughes LE: Nipple retruction in duct ectasia. Br J Surg 64:577-580, 1977 4. Ashworth MT, Corcoran GD, Haqqani MT: Periductal mastitis and mammary duct ectasia in a male. Postgrad Med J 61:621-623, 1985 5. Mansel RE, Morgan WP: Duct ectasia in the male. Br J Surg 66:660-662, 1979 6. Bober E, Ozer E, Akgur F, et al: Bilateral breast masses and
bloody nipple discharge in a two year-old boy. J Pediatr Endocrinol Metab 9:419-421, 1996 7. Stringel G, Perelman A, Jimenez C: Infantile mammary duct ectasia: A cause of bloody nipple discharge. J Pediatr Surg 21:671-674, 1986 8. Bundred NJ, Dixon JM, Lumsden AB, et al: Are the lesions of duct ectasia sterile? Br J Surg 72:844-845, 1985 9. Aitken RJ, Hood J, Going JJ, et al: Bacteriology of mammary duct ectasia. Br J Surg 75:1040-1041, 1988 10. Dixon JM, Anderson TJ, Lumsden AB, et al: Mammary duct ectasia. Br J Surg 70:601-603, 1983
Mammary duct ectasia occurs rarely in childhood. The authors report on the case of a pubertal girl who was operated on for duct ectasia with bloody nipple discharge. Duct ectasia is regarded as a primary lesion; it is considered to be a cause of bloody secretion, and it has a mechanism similar to that of mammary duct papilloma.
J Pediatr Surg 36:E2. Copyright Company.
M
of the above-mentioned evidence, the girl was diagnosed as having a possible case of mammary duct ectasia.
AMMARY DUCT ECTASIA is one of the causes of abnormal nipple discharge, usually found in perimenopausal para, and it rarely occurs in childhood. Its cause is unknown. We observed a case in a 10-yearold girl of mammary duct ectasia. The patient came to the hospital complaining chiefly of bloody nipple discharge. Here we report our findings with a review of the literature. CASE REPORT On September 28, 1998, a premenarcheal girl, 10 years, 8 months old, visited our clinic complaining of bloody discharge from the left nipple. No problem was recognized in her medical history, family history, or development. In the cytology of the secretion, only red blood cells and foam cells were found, and epithelial cells were not recognized. In ultrasonography and computed tomography (CT) scan, marked dilation of the mammary ducts was observed, extending to the periphery from just beneath the nipple, but no clear tumor images were found (Fig 1). There were no abnormal findings in the blood or biochemical test results, blood hormone level, or thyroid function findings. In bacterial cultures of the secreta, Streptococcus spp. were detected on the first occasion and Staphylococcus epidermidis on the second. However, the possibility of bacterial contamination could not be ruled out, because there was no clinical sign of infection. Therefore, antibacterial therapy was not carried out. Because she was in her growth phase, and because no malignancy was observed with cytology, follow-up was carried out. She had her menarche during the follow-up. Because the nipple discharge continued, and no change was seen in to imaging, surgery was performed on July 23, 1999. Arc incision of approximately a semicircle was made in the lower half of the left areola, and extraction of the dilated ducts, along with a little of the breast tissue, was carried out. Microscopic findings (Fig 2) showed that the epithelium of the markedly dilated mammary duct had approximately 2 layers of columnar epithelium and myoepithelial cells. Slight papillary erythrocytes and degenerated necrotic tissue were observed inside the dilated mammary duct, with proliferation in several areas. There was a minor amount of small round cell infiltration in the subepithelium of the mammary duct. Strong hemostasis and fibrotic infiltration were noticed in several parts of the stroma. Hemosiderosis and a small amount of inflammation histiocyte collection were noticed in only one place, but mammary duct damage was not found in this area. In the mammary duct, no remarkable intraluminal obliteration was noticed. On the basis Journal of Pediatric Surgery, Vol 36, No 6 (June), 2001: E2
©
2001 by W.B. Saunders
INDEX WORDS: Mammary duct ectasia, bloody nipple discharge, puberty.
DISCUSSION
The term mammary duct ectasia was proposed by Haagensen (1951)1 for benign lesion of the breast consisting of dilation of the mammary duct, periductal fibrosis, and inflammation, and is accepted widely today. Mammary duct ectasia commonly occurs during perimenopausal multipara,2 but it occurs rarely in nullipara,3 men,4,5 and children.6,7 Regarding mammary duct ectasia of children, no reports of occurrences in pubertal girls are found, although there are reports of occurrences in boys6 and infants.7 In these cases, cause of duct ectasia could not be specified, but the lesion was unilateral. Hormone abnormality was not recognized, and the obstruction finding was not histologically seen in the collecting tubules in the vicinity of the nipple. There are some reports8,9 that bacterial infection is the cause. In this case, bacterial culture of the nipple secretion was positive, but clinical and histologic finding of abscess and other forms of inflammation were lacking. Although the possibility of contamination could not be ruled out, there did not seem to be an association with the cause of disease. Variability of histologic findings of mammary duct ectasia is regarded as depending on the stage of the disease,10 although there is controversy as to which condition is the first development of the inflammatory change of stroma and dilation of mammary duct.2 Dixon et al From the Second Department of Surgery, Department of Pathology, Faculty of Medicine, Toho University, and the Department of Surgery, Kobayashi Hospital, Tokyo, Japan. Address reprint requests to Shinzo Kitahara, Second Department of Surgery, Faculty of Medicine, Toho University, 6-11-1, Omori Nishi, Otaku, Tokyo 143-8541, Japan. Copyright © 2001 by W.B. Saunders Company 1531-5037/01/3606-0040$35.00/0 doi:10.1053/jpsu.2001.24011 1
2
KITAHARA ET AL
Fig 1. Ultrasonographic findings: Mammary ducts in radiating dilation are visualized around nipple.
Fig 2. Histologically, erythrocytes and necrotic tissue are found in the lumen of the mammary duct, which is dilated markedly, and hemosiderin leakage is noticeable in the stroma. The duct epithelium shows papillary growth in some areas.
(1986)10 suggested that periductal mastitis preceded mammary duct ectasia, and that it was the first change in this condition, based on the result that surrounding periductal inflammation of the nondilated duct is marked in younger patients. In older patients, however, marked duct ectasia accompanied by nipple retraction often is observed. However, according to findings to the effect that almost no inflammatory cell infiltration of stroma is found even if mammary duct dilation is markedly revealed and leakage of hemosiderin from mammary duct
to stroma is recognized, it is thought that duct ectasia is the primary lesion. Some areas with papillary growth are found in dilated mammary duct epithelium, and so the mechanism is considered to be a cause of bloody discharge, as in mammary duct papilloma. Considering that the girl was in her growth period, a less invasive method, involving little breast deformation was deemed appropriate. It was thought that a skin incision in the surrounding areola and extirpation of dilated ducts should be satisfactory in this respect.
REFERENCES 1. Haagensen CD: Mammary duct ectasia: A disease that may simulate carcinoma. Cancer 4:749-761, 1951 2. Millis RR: In Gresham GA (ed): Atlas of Breast Pathology vol 7. Lancaster, England, MTP Press Limited, 1984, pp 21-23 3. Rees BI, Gravelle, IH, Hughes LE: Nipple retruction in duct ectasia. Br J Surg 64:577-580, 1977 4. Ashworth MT, Corcoran GD, Haqqani MT: Periductal mastitis and mammary duct ectasia in a male. Postgrad Med J 61:621-623, 1985 5. Mansel RE, Morgan WP: Duct ectasia in the male. Br J Surg 66:660-662, 1979 6. Bober E, Ozer E, Akgur F, et al: Bilateral breast masses and
bloody nipple discharge in a two year-old boy. J Pediatr Endocrinol Metab 9:419-421, 1996 7. Stringel G, Perelman A, Jimenez C: Infantile mammary duct ectasia: A cause of bloody nipple discharge. J Pediatr Surg 21:671-674, 1986 8. Bundred NJ, Dixon JM, Lumsden AB, et al: Are the lesions of duct ectasia sterile? Br J Surg 72:844-845, 1985 9. Aitken RJ, Hood J, Going JJ, et al: Bacteriology of mammary duct ectasia. Br J Surg 75:1040-1041, 1988 10. Dixon JM, Anderson TJ, Lumsden AB, et al: Mammary duct ectasia. Br J Surg 70:601-603, 1983