Rare case of bleeding nipple hemangioma in a lactating mother

Rare case of bleeding nipple hemangioma in a lactating mother Hemangiomas of nipple of the breast are rare lesions and are limited to case reports in the literature. Such pathology in a lactating mother are clearly a hindrance preventing the child from breastfeeding with its benefits. We report a rare case of a bleeding nipple hemangioma in a 24 years old lactating mother with the clinical, imaging and histopathological findings. Before current presentation in our Breast and Thyroid Unit, INTRODUCTION she had presented in emergency for bleeding from the swelling The increasing awareness regarding the breast cancer has led 5 days back which was managed with compression bandaging. the nipple growths to be alarming to the patient. It is therefore The patient came to our Unit for further management. The important for breast surgeons to be able to correctly diagnose patient does not have any co-morbidities including liver disease. these lesions. Hemangiomas of breast are rare benign vascular She is a non-smoker, consumes alcohol occasionally. She lesions without endothelial hyperplasia, atypia and inter- does not have history of malena, hematemesis, epistaxis, hor- anastomosing vascular channels of angiosarcoma [1, 2]. They monal contraceptive use or radiotherapy. She had menarche at can occur in any age group. Though there are no epidemio- the age of 16 years and works as homemaker. logical data on prevalence of benign tumors of nipple, a case The patient is fairly built with no anemia or bruises over the series by Spyropoulou et al. [3] included four benign lesions of body. There was a 2 × 1.5 cm swelling in the superior aspect of nipple. However, none were hemangioma. In this report we left nipple with bluish discoloration of the surface. There was a present a rare case of bleeding nipple hemangioma in a lactat- scab medially, the site of bleeding for which patient had pre- ing mother and review the literature. sented earlier in emergency. The swelling was partially com- pressible. On retro-areolar application of gentle pressure, there was milk discharge from the ducts below the swelling. The CASE REPORT patient did not have axillary lymphadenopathy and signs of A 24-year-old lactating mother from Dhading district of Nepal chronic liver disease. presented with a bluish discolored swelling in her left nipple The patient had normal complete blood count and coagula- for 2 months. The swelling was initially small in size present tion profile. Renal function was normal. Patient was seronega- on the superior aspect of left nipple that grew to a current size tive for HIV, HBV and HCV. over a course of month. She has a 10 months old child and was Ultrasound of the swelling revealed 2 × 1.5 cm well defined having difficulty breastfeeding from left breast. heteroechoic lesion with marked vascularity with both arterial Received: March 28, 2018. Accepted: May 2, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 S. Khanal et al. Figure 1: Photograph showing the lesion on superior aspect of left nipple. Figure 3: Photograph of the cut section after excision of the lesion. Figure 2: Ultrasound image showing the marked vascularity of lesion. Figure 4: Photograph of the nipple after excision. and venous patterns of waveform in superior aspect of left nip- excision of the lesion after discussion of the risks of surgery ple. Rest of the breast, areola and axilla were normal. (possibility of need of excision of nipple should the need arise The diagnosis of vascular lesion was made. Given the risks and consequent affection of breastfeeding). Excision was of re-bleeding and difficulty in breastfeeding, we decided for uneventful. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Rare case of bleeding nipple hemangioma 3 Figure 5: Histopathology showing stratified squamous epithelial lining with proliferation of capillaries in underlying stroma (H & E stain). (a)10× Magnification (b)40× Magnification. The resected specimen was firm, well circumscribed mass ACKNOWLEDGEMENTS with cystic spaces. Dr Anuja Bhandari, Resident, Department of Pathology. Histopathological examination of the lesion showed kerati- nized stratified squamous epithelium with underlying stroma showing proliferation of capillary blood vessels separated by CONFLICT OF INTEREST STATEMENT fibrous connective tissue. The capillaries were lined by bland None of the authors has any conflict of interest to report. epithelial cells. Thus, the diagnosis of capillary hemangioma of nipple was made (Figs 1–5). FUNDING At 3 months follow up, patient was doing fine and was breastfeeding. No funding was received from any source for this work. CONSENT DISCUSSION Written informed consent was obtained for publication of this Though common in other parts of body, there are few reported report and accompanying images. examples of hemangiomas of breast. More so are the capillary hemangiomas of nipple. In the 16 cases of breast hemangiomas studied by Mesurolle et al., 14 were cavernous and only 2 were REFERENCES capillary hemangiomas [4]. There is only a single case of bleed- 1. Smythe FW. Brief communication. Ann Surg 1942;115:716–9. ing hemangioma of nipple in literature in a 16 years old male 2. Rosen PP, Ridolfi RL. The perilobular hemangioma: a benign [5]. Though cavernous hemangiomas of breast are more likely microscopic vascular lesion of the breast. Am J Clin Pathol to present as bleeding, our case is unique in that it was a capil- 1977;68:21–3. lary hemangioma. It can be argued that bleeding could have 3. Spyropoulou G a., Pavlidis L, Trakatelli M, Athanasiou E, been due to trauma of breastfeeding, but it could not be only Pazarli E, Sotiriadis D, et al. Rare benign tumours of the nip- one factor. The rapid increase in size during lactation and the ple. J Eur Acad Dermatol Venereol 2015;29:7–13. increased vascularity during lactation are other factors. Though 4. Mesurolle B, Sygal V, Lalonde L, Lisbona A, Dufresne M-P, florid papillomatosis are the common lesions to present with Gagnon JH, et al. Sonographic and mammographic appear- bleeding from nipple, a high index of suspicion for the possibil- ances of breast hemangioma. Am J Roentgenol 2008;191: ity of hemangioma should be kept in mind [5]. Complete exci- W17–22. sion is recommended for all hemangiomas of breast including 5. Doctor VM, Sirsat MV. Florid papillomatosis (Adenoma) and nipple to exclude the possibility of underlying low grade angio- other benign tumours of the nipple and areola. Br J Cancer sarcoma [6] and due to the difficulty of correct preoperative 1971;25:1–9. diagnosis [7, 8]. The diagnosis of benign vascular tumors of the 6. MD JRH. Lippman ME, MM MD, CKO MD, eds. Diseases of the breast should be achievable based on microscopic findings Breast 5e. 5th edn. Philadelphia: LWW, 2014;1224. alone in most cases. Only in doubt the specimen can be evalu- 7. Funamizu N, Tabei I, Sekine C, Fuke A, Yabe M, Takeyama H, ated for expression of endothelial cell markers (CD31, CD34, et al. Breast hemangioma with difficulty in preoperative factor VIII and Fli-1) [9] which was not required in our case. diagnosis: a case report. World J Surg Oncol [Internet]. 2014 Oct 14 [cited 2018 Mar 26];12. https://www.ncbi.nlm.nih.gov/ pmc/articles/PMC4200173/ CONCLUSION 8. Desbiens C, Hogue J-C, Lévesque Y. Primary breast angiosar- Though rare, hemangiomas of breast and especially the nipple coma: avoiding a common trap [Internet]. Case Reports in can have a significant esthetic and functional concern, and can Oncological Medicine. 2011 [cited 2018 Mar 27]. https://www. increase in size during lactation. Capillary hemangioma should hindawi.com/journals/crionm/2011/517047/ also be considered in bleeding nipple lesions if it grows rela- 9. Folpe AL, Chand EM, Goldblum JR, Weiss SW. Expression of tively rapidly during lactation. Excision is advised to rule out Fli-1, a nuclear transcription factor, distinguishes vascular the possibility of low grade angiosarcoma, facilitate breastfeed- neoplasms from potential mimics. Am J Surg Pathol 2001;25: ing and reduce anxiety. 1061–6. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Rare case of bleeding nipple hemangioma in a lactating mother

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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.
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Abstract

Hemangiomas of nipple of the breast are rare lesions and are limited to case reports in the literature. Such pathology in a lactating mother are clearly a hindrance preventing the child from breastfeeding with its benefits. We report a rare case of a bleeding nipple hemangioma in a 24 years old lactating mother with the clinical, imaging and histopathological findings. Before current presentation in our Breast and Thyroid Unit, INTRODUCTION she had presented in emergency for bleeding from the swelling The increasing awareness regarding the breast cancer has led 5 days back which was managed with compression bandaging. the nipple growths to be alarming to the patient. It is therefore The patient came to our Unit for further management. The important for breast surgeons to be able to correctly diagnose patient does not have any co-morbidities including liver disease. these lesions. Hemangiomas of breast are rare benign vascular She is a non-smoker, consumes alcohol occasionally. She lesions without endothelial hyperplasia, atypia and inter- does not have history of malena, hematemesis, epistaxis, hor- anastomosing vascular channels of angiosarcoma [1, 2]. They monal contraceptive use or radiotherapy. She had menarche at can occur in any age group. Though there are no epidemio- the age of 16 years and works as homemaker. logical data on prevalence of benign tumors of nipple, a case The patient is fairly built with no anemia or bruises over the series by Spyropoulou et al. [3] included four benign lesions of body. There was a 2 × 1.5 cm swelling in the superior aspect of nipple. However, none were hemangioma. In this report we left nipple with bluish discoloration of the surface. There was a present a rare case of bleeding nipple hemangioma in a lactat- scab medially, the site of bleeding for which patient had pre- ing mother and review the literature. sented earlier in emergency. The swelling was partially com- pressible. On retro-areolar application of gentle pressure, there was milk discharge from the ducts below the swelling. The CASE REPORT patient did not have axillary lymphadenopathy and signs of A 24-year-old lactating mother from Dhading district of Nepal chronic liver disease. presented with a bluish discolored swelling in her left nipple The patient had normal complete blood count and coagula- for 2 months. The swelling was initially small in size present tion profile. Renal function was normal. Patient was seronega- on the superior aspect of left nipple that grew to a current size tive for HIV, HBV and HCV. over a course of month. She has a 10 months old child and was Ultrasound of the swelling revealed 2 × 1.5 cm well defined having difficulty breastfeeding from left breast. heteroechoic lesion with marked vascularity with both arterial Received: March 28, 2018. Accepted: May 2, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 S. Khanal et al. Figure 1: Photograph showing the lesion on superior aspect of left nipple. Figure 3: Photograph of the cut section after excision of the lesion. Figure 2: Ultrasound image showing the marked vascularity of lesion. Figure 4: Photograph of the nipple after excision. and venous patterns of waveform in superior aspect of left nip- excision of the lesion after discussion of the risks of surgery ple. Rest of the breast, areola and axilla were normal. (possibility of need of excision of nipple should the need arise The diagnosis of vascular lesion was made. Given the risks and consequent affection of breastfeeding). Excision was of re-bleeding and difficulty in breastfeeding, we decided for uneventful. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Rare case of bleeding nipple hemangioma 3 Figure 5: Histopathology showing stratified squamous epithelial lining with proliferation of capillaries in underlying stroma (H & E stain). (a)10× Magnification (b)40× Magnification. The resected specimen was firm, well circumscribed mass ACKNOWLEDGEMENTS with cystic spaces. Dr Anuja Bhandari, Resident, Department of Pathology. Histopathological examination of the lesion showed kerati- nized stratified squamous epithelium with underlying stroma showing proliferation of capillary blood vessels separated by CONFLICT OF INTEREST STATEMENT fibrous connective tissue. The capillaries were lined by bland None of the authors has any conflict of interest to report. epithelial cells. Thus, the diagnosis of capillary hemangioma of nipple was made (Figs 1–5). FUNDING At 3 months follow up, patient was doing fine and was breastfeeding. No funding was received from any source for this work. CONSENT DISCUSSION Written informed consent was obtained for publication of this Though common in other parts of body, there are few reported report and accompanying images. examples of hemangiomas of breast. More so are the capillary hemangiomas of nipple. In the 16 cases of breast hemangiomas studied by Mesurolle et al., 14 were cavernous and only 2 were REFERENCES capillary hemangiomas [4]. There is only a single case of bleed- 1. Smythe FW. Brief communication. Ann Surg 1942;115:716–9. ing hemangioma of nipple in literature in a 16 years old male 2. Rosen PP, Ridolfi RL. The perilobular hemangioma: a benign [5]. Though cavernous hemangiomas of breast are more likely microscopic vascular lesion of the breast. Am J Clin Pathol to present as bleeding, our case is unique in that it was a capil- 1977;68:21–3. lary hemangioma. It can be argued that bleeding could have 3. Spyropoulou G a., Pavlidis L, Trakatelli M, Athanasiou E, been due to trauma of breastfeeding, but it could not be only Pazarli E, Sotiriadis D, et al. Rare benign tumours of the nip- one factor. The rapid increase in size during lactation and the ple. J Eur Acad Dermatol Venereol 2015;29:7–13. increased vascularity during lactation are other factors. Though 4. Mesurolle B, Sygal V, Lalonde L, Lisbona A, Dufresne M-P, florid papillomatosis are the common lesions to present with Gagnon JH, et al. Sonographic and mammographic appear- bleeding from nipple, a high index of suspicion for the possibil- ances of breast hemangioma. Am J Roentgenol 2008;191: ity of hemangioma should be kept in mind [5]. Complete exci- W17–22. sion is recommended for all hemangiomas of breast including 5. Doctor VM, Sirsat MV. Florid papillomatosis (Adenoma) and nipple to exclude the possibility of underlying low grade angio- other benign tumours of the nipple and areola. Br J Cancer sarcoma [6] and due to the difficulty of correct preoperative 1971;25:1–9. diagnosis [7, 8]. The diagnosis of benign vascular tumors of the 6. MD JRH. Lippman ME, MM MD, CKO MD, eds. Diseases of the breast should be achievable based on microscopic findings Breast 5e. 5th edn. Philadelphia: LWW, 2014;1224. alone in most cases. Only in doubt the specimen can be evalu- 7. Funamizu N, Tabei I, Sekine C, Fuke A, Yabe M, Takeyama H, ated for expression of endothelial cell markers (CD31, CD34, et al. Breast hemangioma with difficulty in preoperative factor VIII and Fli-1) [9] which was not required in our case. diagnosis: a case report. World J Surg Oncol [Internet]. 2014 Oct 14 [cited 2018 Mar 26];12. https://www.ncbi.nlm.nih.gov/ pmc/articles/PMC4200173/ CONCLUSION 8. Desbiens C, Hogue J-C, Lévesque Y. Primary breast angiosar- Though rare, hemangiomas of breast and especially the nipple coma: avoiding a common trap [Internet]. Case Reports in can have a significant esthetic and functional concern, and can Oncological Medicine. 2011 [cited 2018 Mar 27]. https://www. increase in size during lactation. Capillary hemangioma should hindawi.com/journals/crionm/2011/517047/ also be considered in bleeding nipple lesions if it grows rela- 9. Folpe AL, Chand EM, Goldblum JR, Weiss SW. Expression of tively rapidly during lactation. Excision is advised to rule out Fli-1, a nuclear transcription factor, distinguishes vascular the possibility of low grade angiosarcoma, facilitate breastfeed- neoplasms from potential mimics. Am J Surg Pathol 2001;25: ing and reduce anxiety. 1061–6. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy108/5017814 by Ed 'DeepDyve' Gillespie user on 21 June 2018

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Journal of Surgical Case ReportsOxford University Press

Published: May 29, 2018

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