Burkitt's lymphoma of bilateral breasts in pregnancy combined with a huge abdominal mass and bone marrow invasion: A case report

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Abstract

Burkitt's lymphoma of the breast is a rare kind of malignancy, which usually occurs among women during pregnancy or breastfeeding. A rapid diffuse swelling of bilateral breasts is the most common mode of presentation. This article reports a rare case of Burkitt's lymphoma of the breast during pregnancy. The patient presented with diffuse enlargement of both breasts with skin erythema, and increased skin temperature, which brought difficulty in distinguishing it from inflammatory breast cancer and gestational mastitis. The rare clinical manifestations pose a challenge for early diagnosis, only by a series of imaging tests. Until the fine needle aspiration of breast revealed lymphoma cells and a subsequent bone marrow biopsy revealed a translocation of chromosome No.8 and No.14, the final diagnosis was ensured. Meanwhile, imaging examination indicated a huge retroperitoneal mass at a maximum diameter about 10 centimetre. It was significantly reduced together with breast lesion during the standardized chemotherapy. For a rapid progression and poor prognosis, an early diagnosis and prompt therapy are especially important for this very disease. This article provides a patient with an atypical presentation accompanied by a massive retroperitoneal neoplasm, to alert clinicians and help them learn more about this rare disease.
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Burkitt's lymphoma of bilateral breasts in pregnancy combined with a huge abdominal mass and bone marrow invasion: A case report | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Burkitt's lymphoma of bilateral breasts in pregnancy combined with a huge abdominal mass and bone marrow invasion: A case report Xingmin Yan, Shiqiang Liu, Yun Xia This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-3882074/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Burkitt's lymphoma of the breast is a rare kind of malignancy, which usually occurs among women during pregnancy or breastfeeding. A rapid diffuse swelling of bilateral breasts is the most common mode of presentation. This article reports a rare case of Burkitt's lymphoma of the breast during pregnancy. The patient presented with diffuse enlargement of both breasts with skin erythema, and increased skin temperature, which brought difficulty in distinguishing it from inflammatory breast cancer and gestational mastitis. The rare clinical manifestations pose a challenge for early diagnosis, only by a series of imaging tests. Until the fine needle aspiration of breast revealed lymphoma cells and a subsequent bone marrow biopsy revealed a translocation of chromosome No.8 and No.14, the final diagnosis was ensured. Meanwhile, imaging examination indicated a huge retroperitoneal mass at a maximum diameter about 10 centimetre. It was significantly reduced together with breast lesion during the standardized chemotherapy. For a rapid progression and poor prognosis, an early diagnosis and prompt therapy are especially important for this very disease. This article provides a patient with an atypical presentation accompanied by a massive retroperitoneal neoplasm, to alert clinicians and help them learn more about this rare disease. Burkitt's lymphoma Breast neoplasms pregnancy Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 1. Background Lymphoma of the breast is very rare, with a prevalence from 0.5–1% of breast malignancies and 2% of all extra-nodal lymphomas. 1 Burkitt’s breast lymphoma constitutes only about 10% of breast lymphomas and typically happends in pregnant or lactating women, which mostly presents as diffuse bilateral enlargement of breasts. 2 We reported a pregnant woman with physical exam features resembling mastitis and inflammatory breast cancer, but diagnosed with Burkitt's breast lymphoma with a large abdominal mass and bone marrow infiltration. The clinical presentation was highly unusual, which brought a challenge to the diagnosis. 2. Case presentation On May 5, 2023, a 30-year-old gravida at 25 3/7 weeks of gestation was admitted to our department for bilateral enlargement of breasts accompanied by pain. Physical examination disclosed diffusely enlarged, medium-indurated breasts without palpable focal mass. There were extensive erythema, elevated temperature o f skin and increased blood vessels on bilateral breasts (Fig. 1 ). Palpable and matted lymph nodes were found in bilateral axilla and there was no other enlarged lymph nodes found in supra-clavicular, cervical or inguinal. The abdomen was distended, consistent with gestational changes and no limited mass on the abdominal wall were touched. Ultrasound of breast showed markedly thickened glands and diffuse parenchymal disease without focal masses (Fig. 2 ). MRI of breast suggested symmetrically enlarged, dense glands with significant diffusion restriction. In addition, abdominal ultrasound (Fig. 3 ) suggested a 10.1*7.0*8.5 cm hypoechoic area in the right lower abdomen, with irregular echogenicity and abundant blood flow signals. CT of brain and ultrasound of liver, kidney, pancreas, groin and neck were normal. Upon further questioning, the patient complained of pain and swelling in both breasts a month ago. constitutional symptoms including fever, chills, night sweats and weight loss were denied. Recent history of miscarriage or breastfeeding and family history of cancer were not mentioned. An ultrasound at local hospital showed no obvious abnormality, and the primary physician consider it a normal pregnancy reaction. Then, a diagnostic cytological puncture of the right breast was performed, which revealed lymphoma cells (Fig. 4 A). The patient underwent further bone marrow aspiration. Bone marrow cytology revealed a large population of mononuclear cells with dark blue cytoplasm rich in vacuoles (Fig. 4 B).The fluorescence in situ hybridization (FISH) suggested a translocation of chromosome No.8 and No.14, and athology was consistent with the features of Burkitt's lymphoma (BL) (Fig. 5 ). Combining clinical presentation and inspection results, the patient was finally diagnosed with BL, stage Ⅳb. Considering the highly aggressiveness of BL and the advanced stage, the doctors recommended a termination of pregnancy and immediate chemotherapy. The patient has now completed her first chemotherapy treatment. The breast and abdominal ultrasound currently showed no significant masses and tumor cells in bone marrow have disappeared. 3. Discussion BL is a highly aggressive B-cell lymphoma that is common in adolescents but accounts for only 1–2% of adult non-Hodgkin's lymphomas. 3 It is characterized by rapid progression and early hematogenous dissemination with a tendency to metastasize to the bone marrow and central nervous system. 4 Disseminated BL can involve almost any organ, usually presenting as a rapidly growing abdominal mass. Bone marrow is involved in 30–50% of cases, while the involvement ofbreast is extremely rare, accounting for less than 6% of all breast lymphomas. 3 In addition, BLs during pregnancy have a greater tendency to involve the breast with an advanced stage,which may be related to pregnancy-associated immunodeficiencies or the high level of sex hormone stimulation. 5 , 6 One study found that more than 50% of breast BL cases were associated with pregnancy or breastfeeding, and 61.5% of breast BLs in pregnancy were stage IV, 7 which was significantly higher than in non-pregnant lymphoma patients. In this presented case, the initial clinical manifestations and local breast ultrasound results were similar to normal hyperplasia during pregnancy, mostly accounting for the initial missed diagnosis. Besides, signs of the retroperitoneal mass are obscured by an enlarged abdomen in pregnancy, resulting in insufficient clinical evidence for malignancy. This suggests overlapping signs between pregnancy and abdominal lymphoma may lead a delayed diagnosis. And this may also be another reason for an advanced stage of lymphoma in pregnancy at initial diagnosis. The most common symptom of Burkitt's breast lymphoma is diffuse bilateral breast enlargement, 2 and skin recession and erythema are uncommon. This patient uniform breast enlargement with large erythema is really unusual, which complicates the clinical diagnosis. Differential diagnosis includes mastitis and inflammatory breast cancer in patients with reddened breast skin. However, there is a lack of distinct mammographic or ultrasound features to distinguish breast lymphoma from breast carcinoma or benign breast lesions. The finding of a large mass on abdominal ultrasound in this case was suggestive and instructional in considering lymphoma, as inflammatory breast cancer and acute mastitis rarely involve the abdominal lymph nodes. In general, the final diagnosis of breast lymphoma heavily relied on biopsies.At present, systemic chemotherapy is the main treatment of breast lymphoma is. 8 The long-term efficacy of surgery is minimal as this kind of malignancy is sensitive to both chemotherapy and radiotherapy. 1 Meanwhile, the treatment of pregnant woman requires comprehensive consideration according to pregnancy status, tumor aggressiveness, and patient wishes. At one time, it was regarded that once the diagnosis of lymphoma in pregnancy was ensured, labor should be conducted immediately for cytotoxicity of chemical drugs to the fetus. 9 However, no ample evidence prove that labor induction could improve prognosis. 10 Also, studies have shown that the risk of congenital malformations associated with chemotherapy after mid-gestation is close to that of the general population. 10 Therefore, with a close observation and monitoring of both patients and fetuses, continuation of the pregnancy into the middle or late stages of gestation is to be expected. Declarations Ethics approval: Written informed consent was obtained from the individual for the publication of any potentially identifiable images included in this article. Availability of data and materials: The original contributions presented in the study are included in the article/ Supplementary Material. Further inquiries can be directed to the corresponding author. Competing interests: The authors declare that they have no competing interests. Founding: This study has no funding source. Authors' contributions: Yun Xia was responsible for the overall project progress, paper revision, and submission. Xingmin Yan was responsible for writing the paper, organized the image data, and analyzed the data. Shiqiang Liu was responsible for collecting clinicopathological data and revising the manuscript. All authors read and approved the final manuscript. Acknowledgements: None. References Anne N, Pallapothu R. Lymphoma of the breast: a mimic of inflammatory breast cancer. World Journal of Surgical Oncology. 2011;9:125. Hugh JC, Jackson FI, Hanson J, Poppema S. Primary breast lymphoma. An immunohistologic study of 20 new cases. Cancer. 1990;66(12):2602–2611. Roschewski M, Staudt LM, Wilson WH. Burkitt’s Lymphoma. The New England Journal of Medicine. 2022;387(12):1111–1122. Shapira J, Peylan-Ramu N. Burkitt’s lymphoma. Oral Oncology. 1998;34(1):15–23. Horowitz NA, Benyamini N, Wohlfart K, Brenner B, Avivi I. Reproductive organ involvement in non-Hodgkin lymphoma during pregnancy: a systematic review. The Lancet. Oncology. 2013;14(7):e275-282. Levine AM. Lymphoma complicating immunodeficiency disorders. Annals of Oncology: Official Journal of the European Society for Medical Oncology. 1994;5 Suppl 2:29–35. Negahban S, Ahmadi N, Oryan A, Khojasteh HN, Aledavood A, Soleimanpour H, Mohammadianpanah M, Oschlies I, Gesk S, Siebert R, Daneshbod K, Daneshbod Y. Primary bilateral Burkitt lymphoma of the lactating breast: a case report and review of the literature. Molecular Diagnosis & Therapy. 2010;14(4):243–250. Domchek SM, Hecht JL, Fleming MD, Pinkus GS, Canellos GP. Lymphomas of the breast: primary and secondary involvement. Cancer. 2002;94(1):6–13. Hua Z, Ijaz I, Shahzad MN, Yi D, Hu GY, Dong FX. Multidisciplinary Antenatal Management of a Late Pregnancy Complicated With Advanced Stage Breast Burkitt Lymphoma - Case Report and a Review of the Literature. Cureus. 2023;15(2):e34950. Rizack T, Mega A, Legare R, Castillo J. Management of hematological malignancies during pregnancy. American Journal of Hematology. 2009;84(12):830–841. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-3882074","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":268431958,"identity":"ca9cbcc3-ec69-43b4-9449-ff04ac2ba6fc","order_by":0,"name":"Xingmin Yan","email":"","orcid":"","institution":"Huazhong University of Science and Technology","correspondingAuthor":false,"prefix":"","firstName":"Xingmin","middleName":"","lastName":"Yan","suffix":""},{"id":268431959,"identity":"27ce6531-34aa-4b1a-a7a8-cae0400d03c0","order_by":1,"name":"Shiqiang Liu","email":"","orcid":"","institution":"Huazhong University of Science and Technology","correspondingAuthor":false,"prefix":"","firstName":"Shiqiang","middleName":"","lastName":"Liu","suffix":""},{"id":268431960,"identity":"6ea4b1b7-859e-4412-9a00-a8a24cb7f848","order_by":2,"name":"Yun Xia","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA+klEQVRIie3RMUvDQBTA8TsCl+W1WV8IxK/wJNAp0K/ypJDpkIIfwEIgWZq9ix/CRR0jwk1x7ygImToLxRu8ipsm0c3h/sPBHfx4D04In+8fFgWyfrc5XquwNF9vPE7iumyRIec5mMLdaZpQZ9iRglPUi98RsddEDE+XCrq3V7BWRKEmcXwYFnKniR25UmFzn80qEvH2QLLphkmAmlpHZAXPd8lsQ4Lc3EBWw0ShPt98EtR9ApbEcooAmJXgbXHhiEpAuSk4QfD0HbzPMwVmEd9UGWDXrx+bEbJ0KwleY3pWlz0ebJpG9er25ThCvm96Oto/AJ/P5/P90AesQkyRzqGGrgAAAABJRU5ErkJggg==","orcid":"","institution":"Huazhong University of Science and Technology","correspondingAuthor":true,"prefix":"","firstName":"Yun","middleName":"","lastName":"Xia","suffix":""}],"badges":[],"createdAt":"2024-01-20 16:59:13","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-3882074/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-3882074/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":50054972,"identity":"a7d6d3d1-4fb3-4780-9160-b33cae016798","added_by":"auto","created_at":"2024-01-23 17:27:52","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":681527,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eAppearance of both breasts.\u003c/strong\u003e Both breats showed diffuse enlargement with extensive erythema and increased vascularity.\u003c/p\u003e","description":"","filename":"ManuscriptFig1.png","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/f04cbada9b7a44384330c7af.png"},{"id":50054971,"identity":"f5e6423c-2beb-4027-9e2a-c0aaa1539257","added_by":"auto","created_at":"2024-01-23 17:27:52","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":655976,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eUltrasound images of the both breasts\u003c/strong\u003e. Both breasts ultrasound showed markedly swollen and thickened glands, with no limited nodules. A) left breast ultrasound, gland thickness is 7.6cm; B) rightt breast ultrasound, gland thickness is 7.27cm;\u003c/p\u003e","description":"","filename":"ManuscriptFig2.png","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/5c0cf42fc1d48e0510c45747.png"},{"id":50055246,"identity":"a6e3bb1a-6aa6-4d24-82dd-68351dfa9c47","added_by":"auto","created_at":"2024-01-23 17:35:51","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":605487,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eAn ultrasound of the abdomen.\u003c/strong\u003e A hypoechoic mass measuring about 10.33*9.54 cm was seen in the patient's right lower abdomen, with irregular echogenicity and abundant blood flow signals.\u003c/p\u003e","description":"","filename":"ManuscriptFig3.png","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/4b113d91403746c57d93b4fb.png"},{"id":50054969,"identity":"706594f7-6fb5-4413-b36b-ff72c3bd7bb7","added_by":"auto","created_at":"2024-01-23 17:27:51","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":1082770,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eAspiration cytology findings of breast and bone marrow.\u003c/strong\u003eA) fine needle aspiration of the rigth breast (Giemsa stain; x100): a large number of malignant lymphocytes. B)\u003cstrong\u003e \u003c/strong\u003ebone marrow aspiration(Giemsa stain; x400): large groups of single nucleated cells with large cytosol, abundant dark-blue cytoplasm, rich in vacuoles. The arrow points to a lymphoma cell.\u003c/p\u003e","description":"","filename":"ManuscriptFig4.png","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/88dfe1f80bc00d8ad568b7de.png"},{"id":50054973,"identity":"2c767350-d850-46f5-8c67-b2a9bd32cd34","added_by":"auto","created_at":"2024-01-23 17:27:52","extension":"png","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":7276961,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eBiopsy, immunohistochemistry and FISH results of bone marrow tissue. \u003c/strong\u003eA) The biopsy of bone marrow tissue tissue consistent with a diffuse large B cell lymphoma (H\u0026amp;E stain; x40). The arrow points to a B cell. B) The immunostain for CD20, a B-cell marker, is positive (immunoperoxidase stain; x200). In the inset, FISH results revealed that the IGH gene (labeled with a green probe) was fused to the CMYC gene (labeled with a red probe), suggesting the involvement of t(8;14).\u003c/p\u003e","description":"","filename":"ManuscriptFig5.png","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/61eed45d7aebec414b21774f.png"},{"id":50056106,"identity":"015c73b4-136f-4d40-825e-2420262695b7","added_by":"auto","created_at":"2024-01-23 17:51:55","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":5416512,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-3882074/v1/5204fc9f-878c-4c83-88da-88447dbbcaaa.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Burkitt's lymphoma of bilateral breasts in pregnancy combined with a huge abdominal mass and bone marrow invasion: A case report","fulltext":[{"header":"1. Background","content":"\u003cp\u003eLymphoma of the breast is very rare, with a prevalence from 0.5\u0026ndash;1% of breast malignancies and 2% of all extra-nodal lymphomas.\u003csup\u003e\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u003c/sup\u003e Burkitt\u0026rsquo;s breast lymphoma constitutes only about 10% of breast lymphomas and typically happends in pregnant or lactating women, which mostly presents as diffuse bilateral enlargement of breasts.\u003csup\u003e\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e\u003c/sup\u003e We reported a pregnant woman with physical exam features resembling mastitis and inflammatory breast cancer, but diagnosed with Burkitt's breast lymphoma with a large abdominal mass and bone marrow infiltration. The clinical presentation was highly unusual, which brought a challenge to the diagnosis.\u003c/p\u003e"},{"header":"2. Case presentation","content":"\u003cp\u003eOn May 5, 2023, a 30-year-old gravida at 25 3/7 weeks of gestation was admitted to our department for bilateral enlargement of breasts accompanied by pain. Physical examination disclosed diffusely enlarged, medium-indurated breasts without palpable focal mass. There were extensive erythema, elevated temperature o f skin and increased blood vessels on bilateral breasts (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). Palpable and matted lymph nodes were found in bilateral axilla and there was no other enlarged lymph nodes found in supra-clavicular, cervical or inguinal. The abdomen was distended, consistent with gestational changes and no limited mass on the abdominal wall were touched.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eUltrasound of breast showed markedly thickened glands and diffuse parenchymal disease without focal masses (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). MRI of breast suggested symmetrically enlarged, dense glands with significant diffusion restriction. In addition, abdominal ultrasound (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e) suggested a 10.1*7.0*8.5 cm hypoechoic area in the right lower abdomen, with irregular echogenicity and abundant blood flow signals. CT of brain and ultrasound of liver, kidney, pancreas, groin and neck were normal.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eUpon further questioning, the patient complained of pain and swelling in both breasts a month ago. constitutional symptoms including fever, chills, night sweats and weight loss were denied. Recent history of miscarriage or breastfeeding and family history of cancer were not mentioned. An ultrasound at local hospital showed no obvious abnormality, and the primary physician consider it a normal pregnancy reaction.\u003c/p\u003e \u003cp\u003eThen, a diagnostic cytological puncture of the right breast was performed, which revealed lymphoma cells (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003eA). The patient underwent further bone marrow aspiration. Bone marrow cytology revealed a large population of mononuclear cells with dark blue cytoplasm rich in vacuoles (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003eB).The fluorescence in situ hybridization (FISH) suggested a translocation of chromosome No.8 and No.14, and athology was consistent with the features of Burkitt's lymphoma (BL) (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eCombining clinical presentation and inspection results, the patient was finally diagnosed with BL, stage Ⅳb. Considering the highly aggressiveness of BL and the advanced stage, the doctors recommended a termination of pregnancy and immediate chemotherapy. The patient has now completed her first chemotherapy treatment. The breast and abdominal ultrasound currently showed no significant masses and tumor cells in bone marrow have disappeared.\u003c/p\u003e"},{"header":"3. Discussion","content":"\u003cp\u003eBL is a highly aggressive B-cell lymphoma that is common in adolescents but accounts for only 1\u0026ndash;2% of adult non-Hodgkin's lymphomas.\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u003c/sup\u003e It is characterized by rapid progression and early hematogenous dissemination with a tendency to metastasize to the bone marrow and central nervous system.\u003csup\u003e\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e\u003c/sup\u003e Disseminated BL can involve almost any organ, usually presenting as a rapidly growing abdominal mass. Bone marrow is involved in 30\u0026ndash;50% of cases, while the involvement ofbreast is extremely rare, accounting for less than 6% of all breast lymphomas.\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eIn addition, BLs during pregnancy have a greater tendency to involve the breast with an advanced stage,which may be related to pregnancy-associated immunodeficiencies or the high level of sex hormone stimulation.\u003csup\u003e\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e,\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u003c/sup\u003e One study found that more than 50% of breast BL cases were associated with pregnancy or breastfeeding, and 61.5% of breast BLs in pregnancy were stage IV,\u003csup\u003e\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e\u003c/sup\u003e which was significantly higher than in non-pregnant lymphoma patients.\u003c/p\u003e \u003cp\u003eIn this presented case, the initial clinical manifestations and local breast ultrasound results were similar to normal hyperplasia during pregnancy, mostly accounting for the initial missed diagnosis. Besides, signs of the retroperitoneal mass are obscured by an enlarged abdomen in pregnancy, resulting in insufficient clinical evidence for malignancy. This suggests overlapping signs between pregnancy and abdominal lymphoma may lead a delayed diagnosis. And this may also be another reason for an advanced stage of lymphoma in pregnancy at initial diagnosis.\u003c/p\u003e \u003cp\u003eThe most common symptom of Burkitt's breast lymphoma is diffuse bilateral breast enlargement,\u003csup\u003e\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e\u003c/sup\u003e and skin recession and erythema are uncommon. This patient uniform breast enlargement with large erythema is really unusual, which complicates the clinical diagnosis. Differential diagnosis includes mastitis and inflammatory breast cancer in patients with reddened breast skin. However, there is a lack of distinct mammographic or ultrasound features to distinguish breast lymphoma from breast carcinoma or benign breast lesions. The finding of a large mass on abdominal ultrasound in this case was suggestive and instructional in considering lymphoma, as inflammatory breast cancer and acute mastitis rarely involve the abdominal lymph nodes. In general, the final diagnosis of breast lymphoma heavily relied on biopsies.At present, systemic chemotherapy is the main treatment of breast lymphoma is.\u003csup\u003e\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u003c/sup\u003e The long-term efficacy of surgery is minimal as this kind of malignancy is sensitive to both chemotherapy and radiotherapy.\u003csup\u003e\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u003c/sup\u003e Meanwhile, the treatment of pregnant woman requires comprehensive consideration according to pregnancy status, tumor aggressiveness, and patient wishes. At one time, it was regarded that once the diagnosis of lymphoma in pregnancy was ensured, labor should be conducted immediately for cytotoxicity of chemical drugs to the fetus.\u003csup\u003e\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e\u003c/sup\u003e However, no ample evidence prove that labor induction could improve prognosis.\u003csup\u003e\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e\u003c/sup\u003e Also, studies have shown that the risk of congenital malformations associated with chemotherapy after mid-gestation is close to that of the general population.\u003csup\u003e\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e\u003c/sup\u003e Therefore, with a close observation and monitoring of both patients and fetuses, continuation of the pregnancy into the middle or late stages of gestation is to be expected.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the individual for the publication of any potentially identifiable images included in this article.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe original contributions presented in the study are included in the article/ Supplementary Material. Further inquiries can be directed to the corresponding author.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFounding:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study has no funding source.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026apos; contributions:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eYun Xia was responsible for the overall project progress, paper revision, and submission. Xingmin Yan was responsible for writing the paper, organized the image data, and analyzed the data. Shiqiang Liu was responsible for collecting clinicopathological data and revising the manuscript. All authors read and approved the final manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eAnne N, Pallapothu R. Lymphoma of the breast: a mimic of inflammatory breast cancer. World Journal of Surgical Oncology. 2011;9:125.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHugh JC, Jackson FI, Hanson J, Poppema S. Primary breast lymphoma. An immunohistologic study of 20 new cases. Cancer. 1990;66(12):2602\u0026ndash;2611.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRoschewski M, Staudt LM, Wilson WH. Burkitt\u0026rsquo;s Lymphoma. The New England Journal of Medicine. 2022;387(12):1111\u0026ndash;1122.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShapira J, Peylan-Ramu N. Burkitt\u0026rsquo;s lymphoma. Oral Oncology. 1998;34(1):15\u0026ndash;23.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHorowitz NA, Benyamini N, Wohlfart K, Brenner B, Avivi I. Reproductive organ involvement in non-Hodgkin lymphoma during pregnancy: a systematic review. The Lancet. Oncology. 2013;14(7):e275-282.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLevine AM. Lymphoma complicating immunodeficiency disorders. Annals of Oncology: Official Journal of the European Society for Medical Oncology. 1994;5 Suppl 2:29\u0026ndash;35.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eNegahban S, Ahmadi N, Oryan A, Khojasteh HN, Aledavood A, Soleimanpour H, Mohammadianpanah M, Oschlies I, Gesk S, Siebert R, Daneshbod K, Daneshbod Y. Primary bilateral Burkitt lymphoma of the lactating breast: a case report and review of the literature. Molecular Diagnosis \u0026amp; Therapy. 2010;14(4):243\u0026ndash;250.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDomchek SM, Hecht JL, Fleming MD, Pinkus GS, Canellos GP. Lymphomas of the breast: primary and secondary involvement. Cancer. 2002;94(1):6\u0026ndash;13.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHua Z, Ijaz I, Shahzad MN, Yi D, Hu GY, Dong FX. Multidisciplinary Antenatal Management of a Late Pregnancy Complicated With Advanced Stage Breast Burkitt Lymphoma - Case Report and a Review of the Literature. Cureus. 2023;15(2):e34950.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRizack T, Mega A, Legare R, Castillo J. Management of hematological malignancies during pregnancy. American Journal of Hematology. 2009;84(12):830\u0026ndash;841.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Burkitt's lymphoma, Breast neoplasms, pregnancy","lastPublishedDoi":"10.21203/rs.3.rs-3882074/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-3882074/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eBurkitt's lymphoma of the breast is a rare kind of malignancy, which usually occurs among women during pregnancy or breastfeeding. A rapid diffuse swelling of bilateral breasts is the most common mode of presentation. This article reports a rare case of Burkitt's lymphoma of the breast during pregnancy. The patient presented with diffuse enlargement of both breasts with skin erythema, and increased skin temperature, which brought difficulty in distinguishing it from inflammatory breast cancer and gestational mastitis. The rare clinical manifestations pose a challenge for early diagnosis, only by a series of imaging tests. Until the fine needle aspiration of breast revealed lymphoma cells and a subsequent bone marrow biopsy revealed a translocation of chromosome No.8 and No.14, the final diagnosis was ensured. Meanwhile, imaging examination indicated a huge retroperitoneal mass at a maximum diameter about 10 centimetre. It was significantly reduced together with breast lesion during the standardized chemotherapy. For a rapid progression and poor prognosis, an early diagnosis and prompt therapy are especially important for this very disease. This article provides a patient with an atypical presentation accompanied by a massive retroperitoneal neoplasm, to alert clinicians and help them learn more about this rare disease.\u003c/p\u003e","manuscriptTitle":"Burkitt's lymphoma of bilateral breasts in pregnancy combined with a huge abdominal mass and bone marrow invasion: A case report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-01-23 17:27:47","doi":"10.21203/rs.3.rs-3882074/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"cb4f58b9-549e-4e51-97cf-15c5c8f3f6b8","owner":[],"postedDate":"January 23rd, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-01-23T17:27:49+00:00","versionOfRecord":[],"versionCreatedAt":"2024-01-23 17:27:47","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-3882074","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-3882074","identity":"rs-3882074","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

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