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Wang G, Ma X, Wei W, Yao W, Cheng F. Clear cell hidradenoma of the male breast: A case report and literature review. Int J Surg Case Rep 2025; 127:110887. [PMID: 39813983 PMCID: PMC11783126 DOI: 10.1016/j.ijscr.2025.110887] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/20/2024] [Revised: 01/08/2025] [Accepted: 01/11/2025] [Indexed: 01/18/2025] Open
Abstract
INTRODUCTION Clear cell hidradenoma (CCH) is a rare type of benign breast tumor that may undergo malignant transformation in a few cases. It clinically manifests as a painless breast mass, and may include nipple discharge. Complete surgical excision with adequate safety margins is the primary treatment. PRESENTATION OF CASE A 34-year-old man developed a mass beneath the left nipple over 2 years. Although the mass was painless, it gradually enlarged over time and was accompanied by nipple discharge. Complete excision of the mass revealed that the tumor cells were arranged in solid nodule-like structures composed of clear cells and few cells with eosinophilic cytoplasm. Based on the results of immunohistochemical staining and MAML2 rearrangement analysis, a pathological diagnosis of breast CCH was made. The patient was followed up postoperatively for six months, during which there was no recurrence. DISCUSSION Breast CCH differentiates from low-grade mucoepidermoid, glycogen-rich, and lipid-rich breast carcinomas. A more comprehensive understanding of breast CCH can aid pathologists in avoiding misdiagnoses during fine-needle aspiration biopsy and intraoperative frozen section diagnosis. Diagnostic clues for CCH include: (1) the presence of two types of cell morphology in the tumor, (2) immunohistochemical staining indicating p63 positivity, (3) negativity for myoepithelial markers, and (4) MAML2 gene rearrangement in certain cases. CONCLUSION The case we reported occurred in a male patient with MAML2 gene rearrangement, which is extremely rare. Complete surgical excision with adequate safety margins is the primary treatment method for breast CCH, and close postoperative follow-up should be conducted to monitor disease recurrence.
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Affiliation(s)
- Guihua Wang
- Department of Pathology, Liangzhu Branch, The First Affiliated Hospital of Zhejiang University School of Medicine, No.1260 Kangliang Street, Liangzhu Street, Yuhang District, Hangzhou City, Zhejiang Province 311112, China
| | - Xinyi Ma
- Pathological Diagnosis Center of DiAn Diagnostic Technology Group Co., LTD., No.329 Jinpeng Street, Sandun Town, Xihu District, Hangzhou City, Zhejiang Province 310030, China
| | - Wenbing Wei
- Department of Ultrasound Medicine, Binzhou People's Hospital, No.515, Huanghe Seventh Road, Bincheng District, Binzhou City, Shandong Province 256600, China
| | - Wanqi Yao
- Department of Pathology, Liangzhu Branch, The First Affiliated Hospital of Zhejiang University School of Medicine, No.1260 Kangliang Street, Liangzhu Street, Yuhang District, Hangzhou City, Zhejiang Province 311112, China
| | - Fei Cheng
- Department of Pathology, The First Affiliated Hospital of Zhejiang University School of Medicine, No.79 Qingchun Road, Hangzhou, Zhejiang 310003, China.
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Luo L, Hu Y. Clear cell hidradenoma of the breast with MAML2 gene rearrangement. Pathol Int 2024; 74:482-488. [PMID: 38818886 PMCID: PMC11551824 DOI: 10.1111/pin.13455] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/20/2023] [Revised: 05/14/2024] [Accepted: 05/15/2024] [Indexed: 06/01/2024]
Abstract
Clear cell hidradenoma is a rare benign tumor of the breast, its origin and pathogenesis are controversial. We have experienced a case of breast clear cell hidradenoma with mastermind like transcriptional coactivator 2 (MAML2) gene rearrangement. The patient found a painless mass with a hard texture in the left breast areola without nipple discharge. Microscopically, the tumor was cystic and solid, locally arranged in a glandular structure, covered by single cuboidal cells; it was composed of clear cells, epidermoid cells, and basaloid cells; there were no necrosis or mitotic figures. Immunohistochemical staining showed that the tumor cells positively expressed low-molecular cytokeratin 7, low-molecular cytokeratins (Cam5.2), high-molecular cytokeratin 5/6, cytokeratin 14, CD117, and p63; and did not express calponin, and smooth muscle myosin heavy chain. The cuboidal cells were positive for SOX10 but negative for p63. Additionally, periodic acid-Schiff reaction showed purple-red granules in the tumor cytoplasm, but Alcian blue staining showed no blue mucus in the cytoplasm. The split signals of MAML2 gene were detected by fluorescence in situ hybridization. Subtle histological and immunophenotypical differences may help to distinguish breast clear cell hidradenoma from common breast tumors. Furthermore, the MAML2 gene rearrangement may be a molecular genetic characteristic of breast clear cell hidradenoma.
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Affiliation(s)
- Li Luo
- Department of Pathology, Beijing Luhe HospitalCapital Medical UniversityBeijingChina
| | - Yanping Hu
- Department of Pathology, Beijing Luhe HospitalCapital Medical UniversityBeijingChina
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Siluvai Arulappan LA, Eleza BF. Remote Possibility of Post Aural Swelling as Nodular Hidradenoma: A Case Report. Indian J Otolaryngol Head Neck Surg 2024; 76:94-96. [PMID: 38440531 PMCID: PMC10908938 DOI: 10.1007/s12070-023-04089-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/30/2023] [Accepted: 07/13/2023] [Indexed: 03/06/2024] Open
Abstract
A 30-year-old female patient presented with swelling over the left post aural region of 6 months duration which gradually increased in size. Cutaneous examination: A solitary, non-tender, mobile swelling of variable consistency over the post aural region. Dermoscopic examination: Revealed white and red areas. Diagnosis of nodular hidradenoma was made by histopathological examination.
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Affiliation(s)
| | - Blessed Finney Eleza
- Upgraded Institute of Otorhinolaryngology Madras Medical College, Chennai, India
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4
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Panigrahi C, Adhya AK. Cytomorphology of an uncommon cutaneous tumor. Cytopathology 2022; 33:412-414. [PMID: 35191102 DOI: 10.1111/cyt.13112] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/05/2022] [Revised: 01/28/2022] [Accepted: 02/16/2022] [Indexed: 11/30/2022]
Affiliation(s)
- Chinmayee Panigrahi
- Department of Pathology and Laboratory Medicine, All India institute of Medical sciences, Bhubaneswar, Odisha, India
| | - Amit Kumar Adhya
- Department of Pathology and Laboratory Medicine, All India institute of Medical sciences, Bhubaneswar, Odisha, India
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Al-Adawi SSH, Al-Qasabi B, Al-Bahri M, Aljarrah A, Al-Aghbari S. Clear Cell Hidradenoma of the Breast Diagnosed on a Core Needle Biopsy: A case report and review of the literature. Sultan Qaboos Univ Med J 2020; 20:e357-e361. [PMID: 33110653 PMCID: PMC7574794 DOI: 10.18295/squmj.2020.20.03.017] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/03/2020] [Revised: 03/30/2020] [Accepted: 04/27/2020] [Indexed: 11/28/2022] Open
Abstract
Clear cell hidradenoma (CCH) is a tumour originating from the eccrine sweat glands. It usually presents in the limbs, axilla or trunk. CCH of the breast is rare and can present as a cystic lesion in the breast that can be easily misdiagnosed as malignancy. We report a 36-year-old female patient who presented at the Sultan Qaboos University Hospital Breast Clinic, Muscat, Oman, in 2018 with a lump in her left breast. Ultrasound examination reported a complex cystic lesion with a solid, vascular component. An ultrasound-guided core needle biopsy was suggestive of clear cell hidradenoma. Surgical excision was performed and histopathology confirmed the diagnosis of CCH of the breast. This is the first ever case of a diagnosis of CCH made using core needle biopsy. CCH can be challenging to diagnose; therefore, awareness of its histopathological and ultrasonographic features are essential to avoid misdiagnosis and over treatment.
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Affiliation(s)
- Sara S H Al-Adawi
- Department of General Surgery, Oman Medical Speciality Board, Muscat, Oman
| | - Badriya Al-Qasabi
- Department of Radiology, Sultan Qaboos University Hospital, Muscat, Oman
| | - Maiya Al-Bahri
- Department of Pathology, Sultan Qaboos University Hospital, Muscat, Oman
| | - Adil Aljarrah
- Department of General Surgery, Sultan Qaboos University Hospital, Muscat, Oman
| | - Suad Al-Aghbari
- Department of General Surgery, Sultan Qaboos University Hospital, Muscat, Oman
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Ham T, Cheon SJ, Roh MS, Ha DH. Ultrasonography of Malignant Clear Cell Hidradenoma: A Case Report. JOURNAL OF THE KOREAN SOCIETY OF RADIOLOGY 2020; 81:448-452. [PMID: 36237377 PMCID: PMC9431819 DOI: 10.3348/jksr.2020.81.2.448] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 05/29/2018] [Revised: 10/28/2018] [Accepted: 09/02/2019] [Indexed: 11/15/2022]
Abstract
Clear cell hidradenoma is a tumor that originates from a sweat gland and typically involves the dermis and subcutaneous tissue. Malignant clear cell hidradenoma is very rare, and surgical excision is usually performed without imaging. There are few reports of the ultrasonographic findings of malignant clear cell hidradenomas. Herein, we present the ultrasonographic characteristics of a malignant clear cell hidradenoma.
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Affiliation(s)
- Taehyuk Ham
- Department of Radiology, Dong-A University Hospital, Busan, Korea
| | - Sang-jin Cheon
- Department of Dermatology, Yangsan Pusan National University Hospital, Yangsan, Korea
| | - Mee Sook Roh
- Department of Patholgy, Dong-A University Hospital, Busan, Korea
| | - Dong-ho Ha
- Department of Radiology, Dong-A University Hospital, Busan, Korea
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Jaitly V, Jahan-Tigh R, Belousova T, Zhu H, Brown R, Saluja K. Case Report and Literature Review of Nodular Hiradenoma, a Rare Adnexal Tumor That Mimics Breast Carcinoma, in a 20-Year-Old Woman. Lab Med 2019; 50:320-325. [PMID: 30722042 DOI: 10.1093/labmed/lmy084] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/25/2018] [Revised: 11/05/2018] [Accepted: 01/03/2019] [Indexed: 12/29/2022] Open
Abstract
Nodular hidradenoma is an uncommon cutaneous adnexal tumor arising from sweat glands. In the skin, it usually presents as a solitary dermal nodule; excision is curative in most cases. In rare instances, it may present as a breast mass and can mimic breast carcinoma clinically and radiologically, causing diagnostic dilemmas for the treating physician and pathologist. Herein, we discuss a case of nodular hidradenoma in a 20-year-old Hispanic woman as a rapidly growing mass in the breast that mimicked breast carcinoma. We discuss the rare presentation of this uncommon tumor and the differential diagnosis of this entity, as well as the results of our literature review on the topic.
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Affiliation(s)
- Vanya Jaitly
- Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
| | - Richard Jahan-Tigh
- Department of Dermatology, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
| | - Tatiana Belousova
- Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
| | - Hui Zhu
- Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
| | - Robert Brown
- Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
| | - Karan Saluja
- Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center McGovern Medical School, Houston, Texas
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An JK, Woo JJ, Hong YO. Malignant sweat gland tumor of breast arising in pre-existing benign tumor: A case report. World J Clin Cases 2019; 7:3033-3038. [PMID: 31624751 PMCID: PMC6795723 DOI: 10.12998/wjcc.v7.i19.3033] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/27/2019] [Revised: 08/22/2019] [Accepted: 09/09/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Sweat glands belong to skin appendages. Sweat gland tumors are uncommon, especially when they occur as malignant tumors in the breast. We report a case of malignant sweat gland tumor of the breast, including imaging and pathological findings.
CASE SUMMARY A 47-year-old woman visited our hospital with a non-tender palpable lesion in her left breast. The lesion had not shown changes for 10 years. However, it recently increased in size. Sonography showed a well circumscribed cystic lesion with internal debris and fluid-fluid level. Magnetic resonance imaging showed a well circumscribed oval mass with T1 hyper-intensity compared to muscle and T2 high signal intensity. There was a small enhancing mural component in the inner wall of the mass. The tumor was resected. Its pathologic result was a malignant transformation of benign sweat gland tumor such as hidradenoma. The lesion was treated with excision and radiation therapy. At 1-year follow up, there was no local recurrence or metastasis in the patient.
CONCLUSION In the case of a rapid growing cystic mass in the nipple and subareola, it is necessary to distinguish it from a malignant sweat gland tumor.
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Affiliation(s)
- Jin Kyung An
- Department of Radiology, Nowon Eulji Medical Center, Eulji University, Seoul 01830, South Korea
| | - Jeong Joo Woo
- Department of Radiology, Nowon Eulji Medical Center, Eulji University, Seoul 01830, South Korea
| | - Young Ok Hong
- Department of Pathology, Nowon Eulji Medical Center, Eulji University, Seoul 01830, South Korea
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Ano-Edward G, Amole I, Adesina S, Ajiboye O, lasisi M, Jooda R. Nodular hidradenoma of the breast: A case report. ALEXANDRIA JOURNAL OF MEDICINE 2019. [DOI: 10.1016/j.ajme.2018.04.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
Affiliation(s)
- G.H. Ano-Edward
- Department of Anatomic Pathology, College of Health Sciences, Bowen University, Iwo, Nigeria
| | - I.O. Amole
- Department of Family Medicine, College of Health Sciences, Bowen University, Iwo, Nigeria
| | - S.A. Adesina
- Department of Family Medicine, College of Health Sciences, Bowen University, Iwo, Nigeria
| | - O.A. Ajiboye
- Department of Surgery, College of Health Sciences, Bowen University, Iwo, Nigeria
| | - M.E. lasisi
- Department of Anatomic Pathology, College of Health Sciences, Bowen University, Iwo, Nigeria
| | - R.K. Jooda
- Department of Anatomic Pathology, College of Health Sciences, Bowen University, Iwo, Nigeria
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Shalini B, Akshi K, Namrata N, Shilpi S, Manupriya N. Case of chondroid syringoma mimicking a nodular hidradenoma: A diagnostic pitfall on cytopathology. Diagn Cytopathol 2017; 46:59-62. [PMID: 28772003 DOI: 10.1002/dc.23791] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/18/2017] [Revised: 06/27/2017] [Accepted: 07/05/2017] [Indexed: 11/08/2022]
Abstract
Limited literature is available on the use of fine needle aspiration (FNA) for skin tumors. Awareness of cytological features of these lesions is important to prevent misdiagnosis. A 45-year-old male, presented with slowly growing swelling over the thigh since 4 years. FNA from multiple sites yielded highly cellular smears with a dual cell population consisting of cells with small round nucleus scanty cytoplasm and polygonal cells with moderate to abundant cytoplasm. Extracellular basement membrane like material was also noted. A diagnosis of benign adnexal neoplasm, suggestive of nodular hidradenoma was made. Paraffin sections showed an encapsulated dermal tumor composed of ductal and myoepithelial cells in ducts, sheets, strands with chondromyxoid stroma. A final diagnosis of chondroid syringoma was given following evaluation of histopathology slides and Immunohistochemistry. FNA interpretation of skin tumors can be challenging since literature is limited. The placement of tumors into benign or malignant category is realistic and can direct management guidelines. Greater awareness of these lesions and following stringent diagnostic criteria can help in establishing a more accurate diagnosis.
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Affiliation(s)
- Bahadur Shalini
- Department of Pathology, North Delhi Municipal Corporation Medical College and Hindu Rao Hospital, New Delhi, India
| | - Katyal Akshi
- Department of Pathology, North Delhi Municipal Corporation Medical College and Hindu Rao Hospital, New Delhi, India
| | - Nargotra Namrata
- Department of Pathology, North Delhi Municipal Corporation Medical College and Hindu Rao Hospital, New Delhi, India
| | - Soni Shilpi
- Department of Pathology, North Delhi Municipal Corporation Medical College and Hindu Rao Hospital, New Delhi, India
| | - Nain Manupriya
- Department of Pathology, North Delhi Municipal Corporation Medical College and Hindu Rao Hospital, New Delhi, India
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Rooper LM, Iding JS, Cuda JD, Ali SZ. Atypical Hidradenoma Mimicking Primary Mammary Carcinoma on Breast Fine-Needle Aspiration: A Case Report with Long-Term Follow-Up. Acta Cytol 2016; 60:173-8. [PMID: 27010690 DOI: 10.1159/000445095] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/02/2016] [Accepted: 02/29/2016] [Indexed: 12/29/2022]
Abstract
BACKGROUND Fine-needle aspiration (FNA) is well-established as an accurate technique for the diagnosis of palpable breast masses. While the differential diagnosis of such lesions usually focuses on benign or malignant mammary proliferations, a subset of breast neoplasms arises from skin and soft tissue. Skin adnexal neoplasms such as hidradenoma can pose a particular pitfall on breast FNA cytology (FNAC) as epithelial proliferations that are not of ductal or lobular origin. CASE A 59-year-old female presented with a superficial breast mass. FNAC revealed a hypercellular lesion with marked nuclear atypia that was highly suspicious for carcinoma. However, at partial mastectomy, the histological features of the tumor were consistent with atypical hidradenoma. Negativity for estrogen receptor (ER) and progesterone receptor (PR) confirmed the diagnosis. Eighteen years later, the patient remains free of recurrence or metastasis. CONCLUSIONS This report offers the first description of findings of atypical hidradenoma on FNAC. In the breast, its high cellularity and nuclear atypia can mimic a primary mammary carcinoma on FNAC. Although breast and skin adnexal tumors show a immunohistochemical overlap, negative ER and PR stains in a low-grade tumor can suggest a nonmammary lesion. Skin adnexal neoplasms should be considered in the differential diagnosis of superficial breast tumors.
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Affiliation(s)
- Lisa M Rooper
- Department of Pathology, The Johns Hopkins University School of Medicine, Baltimore, Md., USA
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Sehgal S, Goyal P, Ghosh S, Mittal D, Kumar A, Singh S. Clear cell hidradenoma of breast mimicking atypical breast lesion: a diagnostic pitfall in breast cytology. Rare Tumors 2014; 6:5343. [PMID: 25002948 PMCID: PMC4083668 DOI: 10.4081/rt.2014.5343] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/06/2014] [Revised: 03/20/2014] [Accepted: 03/20/2014] [Indexed: 12/11/2022] Open
Abstract
Clear cell hidradenoma (CCH) is an uncommon skin adnexal tumor arising from eccrine glands. Although several kind of skin adnexal tumors arise in the breast tissue, CCH of the breast is an extremely rare entity. Failure to identify its cytomorphologic features and rarity of this tumor may lead to misdiagnosis on fine needle aspiration cytology. Hereby we report a case of 30-year-old female who presented with painless lump in left breast since 10 months. Fine needle aspiration cytology of lump yielded fluid material. On May-Grunwald-Giemsa stained smears, a possibility of atypical breast lesion was considered and patient was advised a biopsy examination. Final diagnosis of CCH was made on histopathologic examination. Awareness of cytomorphologic features of breast CCH will prevent misdiagnosis as malignant or atypical breast lesions and will allow for correct management of the patients.
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Affiliation(s)
- Shelly Sehgal
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
| | - Prashant Goyal
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
| | - Soumyesh Ghosh
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
| | - Deepti Mittal
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
| | - Awanindra Kumar
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
| | - Sompal Singh
- Department of Pathology, Swami Dayanand Hospital , Delhi, India
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