Primary cutaneous anaplastic large-cell lymphoma resembling space infection: a case report

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Background: Primary cutaneous anaplastic large-cell lymphoma (PC-ALCL) is a rare T-cell lymphoma belonging to the CD30+ T-cell lymphoproliferative disorders. The case of PC-ALCL in the temporal and parotid gland region is exceedingly rare. Herein, we report a case of PC-ALCL involving the temporal and parotid gland region mimicking maxillofacial space infection. Case presentation: A 78-year-old woman presented to maxillofacial surgery service with a 6-month history of swelling and pain in the left side of her face. The patient underwent incision and drainage, and we unexpectedly found no pus in the lesion area. Incisional biopsy showed necrosis and extensive involvement of the left temples by a diffuse infiltrate containing large, atypical cells. The tumor cells were positive for CD30, CD3, and Ki67, and negative for ALK (SP8). After considering these findings, a diagnosis of PC-ALCL was rendered. The patient was admitted to the lymphoma department for systemic chemotherapy and no relapse occurred during a follow-up period of three months. Conclusions: : This report suggests that if there are suspicious intraoperative manifestations, carrying out a biopsy simultaneously, using HE staining, comprehensive IHC panels, and combining them with a systematic approach is essential to diagnosing ALCL to prevent misdiagnosis.
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Primary cutaneous anaplastic large-cell lymphoma resembling space infection: 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 Primary cutaneous anaplastic large-cell lymphoma resembling space infection: a case report Xiaohan Yan, Wenhao Ren, Shaoming Li, Zhuang Zhu, Ling Gao, Keqian Zhi This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-3855822/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 8 You are reading this latest preprint version Abstract Background: Primary cutaneous anaplastic large-cell lymphoma (PC-ALCL) is a rare T-cell lymphoma belonging to the CD30+ T-cell lymphoproliferative disorders. The case of PC-ALCL in the temporal and parotid gland region is exceedingly rare. Herein, we report a case of PC-ALCL involving the temporal and parotid gland region mimicking maxillofacial space infection. Case presentation: A 78-year-old woman presented to maxillofacial surgery service with a 6-month history of swelling and pain in the left side of her face. The patient underwent incision and drainage, and we unexpectedly found no pus in the lesion area. Incisional biopsy showed necrosis and extensive involvement of the left temples by a diffuse infiltrate containing large, atypical cells. The tumor cells were positive for CD30, CD3, and Ki67, and negative for ALK (SP8). After considering these findings, a diagnosis of PC-ALCL was rendered. The patient was admitted to the lymphoma department for systemic chemotherapy and no relapse occurred during a follow-up period of three months. Conclusions: This report suggests that if there are suspicious intraoperative manifestations, carrying out a biopsy simultaneously, using HE staining, comprehensive IHC panels, and combining them with a systematic approach is essential to diagnosing ALCL to prevent misdiagnosis. Primary cutaneous anaplastic large-cell lymphoma space infection temporal and parotid gland region case report Figures Figure 1 Figure 2 Figure 3 Background Anaplastic large cell lymphoma (ALCL) is a rare type of non-Hodgkin's lymphoma (NHL), as well as one of the subtypes of T cell lymphoma with ample cytoplasm and pleomorphic nuclei, expressing near-universal levels of the CD30[1, 2]. According to the 2016 WHO classification, there are four types of ALCL: systemic ALK-positive ALCL (ALK+ ALCL), systemic ALK-negative ALCL (ALK− ALCL), primary cutaneous ALCL (PC-ALCL), and breast implant-associated ALCL (BI-ALCL)[3].  PC-ALCL presents on the skin and is characterized by a very good prognosis and response to treatment in the majority of cases. Early diagnosis of PC-ALCL is crucial because the clinical stage is directly related to the prognosis. Considering variations in morphology and immunophenotype, many differential diagnoses should be regarded as due to atypical lymphoma presentations. Here, we present a case of ALCL in a 78-year-old woman with temple and parotid gland region involvement, first thought to be a maxillofacial space infection. Therefore, we summarized some patient history experiences, specialty examination, clinical manifestations, and treatment, hoping to provide essential clues for diagnosing PC-ALCL with first symptoms in rare sites. Case presentation A 78-year-old woman presented to maxillofacial surgery service with a 6-month history of swelling and pain in the left side of her face, which got worse in the past 20 days. 6 months ago, a biopsy was performed at another hospital, and the diagnosis was a chronic inflammatory. The patient had a 5-year history of diabetes mellitus. Clinical examination revealed diffused left-sided temporal swelling and unobvious left parotid swelling, both with tough texture, obvious tenderness, and poor activity. Laboratory investigations found an elevated C-reactive protein (CRP) concentration of 109 ·97 mg/dl and a mildly raised procalcitonin concentration of 0.08 ng/ml. A cone beam computed tomography (CBCT) showed enlarged lymph nodes of levels I to II in the left neck, extensive thickening of subcutaneous tissue of the left temples [Fig.1], and a parotid hypodense lesion together with a blurred image of masticator, buccal, and submandibular space. Considering these findings, we suspected an infratemporal space infection. Based on these, an incision and drainage were planned for the patient. However, we found the lesion area with only a small amount of clear fluid rather than pus. 4 days later, the pain reappeared in the lesion area. The definitive histopathological examination showed necrosis and extensive involvement of the left temples by a diffuse infiltrate containing large, atypical cells [Fig.2a, b]. Immunohistochemical stains showed that the cells were positive for CD30[Fig.3a], CD3[Fig.3b], Ki67[Fig.3c], Bcl-6, MUM1, c-Myc, p53, and EBER. They were negative for ALK (SP8) [Fig.3d], CD20, CD19, CD10, Bcl-2, CyclinD1, CD21, CD5, EBER(P), CD56, Pax-5, CD138, CD8 and CD4. To ensure no metastasis, a positron emission tomography (PET) scan was conducted, and no suspicious metastasis was found. Considering the findings, the diagnosis of PC-ALCL was made. The patient was admitted to the lymphoma department for systemic chemotherapy with CDOP (cyclophosphamide, vindesine, liposome, dexamethasone, and prednisone), and the patient’s clinical status improved after the first treatment cycle. Discussion and conclusions PC-ALCL is a rare ALK-negative T-cell neoplasm with an annual incidence of 10 cases per million individuals[4]. It is commonly seen in people between 50 and 70, but it has also been reported in pediatric cases[5, 6]. Around 80% of cases present with a localized cutaneous nodule or papule, and aggressive extracutaneous dissemination occurs in only about 10% of cases[7-9]. In this clinical case, the lesion occurring in the temple and parotid gland shows diffuse swelling with cumulative regional lymph nodes, combined with the history of diabetes mellitus and misdiagnosis of the first biopsy; all these factors associated with the clinical aspect of the lesion led us to the initially suspected of infratemporal space infection. In the diagnosis of infratemporal space infection, many diseases must be considered for differential diagnosis, such as temporomandibular joint disorder, lymphadenitis, and lymphoma[10]. We speculate that the patient’s pain relief temporarily after incision and drainage may be a decrease in local tension. In addition, clinical conditions do not resolve after the conventional treatment; other possibilities should be considered. Noteworthily, the diagnosis of ALCL requires complex interdisciplinary information integration by clinicians and pathologists[11]. Suppose no pus is found during the operation, we recommend carrying out a biopsy simultaneously, using HE staining, comprehensive IHC panels, and combining them with a systematic approach to diagnosing ALCL to prevent misdiagnosis. Abbreviations PC-ALCL Primary cutaneous anaplastic large-cell lymphoma ALCL Anaplastic large cell lymphoma CRP C-reactive protein CBCT A cone beam computed tomography PET positron emission tomography Declarations Acknowledgements Not applicable. Author contributions Xiaohan Yan performed drafting of the manuscript, writing, and literature search. Xiaohan Yan, Ling Gao, and Keqian Zhi contributed substantially to the conception, design, and patient data acquisition of the manuscript. Wenhao Ren and Shaoming Li performed the biopsy and was involved in revising the manuscript with respect to intellectual content. Zhuang Zhu performed the histopathological diagnosis and contributed to the interpretation of the data. All authors reviewed and approved the final manuscript prior to submission. Ethics approval and consent to participate Not applicable. Consent for publication Written informed consent was obtained from the patient for publication of her clinical details and any accompanying images. A copy of the written consent is available on request. All of the data were anonymized. Competing interests The authors declare that they have no competing interests. Financial support This study was funded by TaiShan Scholars Foundation of Shandong Province (No. tsqn202306397 [LG]). References Pletneva MA, Smith LB. Anaplastic large cell lymphoma: features presenting diagnostic challenges. Arch Pathol Lab Med . 2014;138(10):1290-4.doi.org/10.5858/arpa.2014-0295-CC. Zhang XR, Chien PN, Nam SY, Heo CY. Anaplastic Large Cell Lymphoma: Molecular Pathogenesis and Treatment. Cancers (Basel) . 2022;14(7).doi.org/10.3390/cancers14071650. Swerdlow SH, Campo E, Pileri SA, Harris NL, Stein H, Siebert R, et al. The 2016 revision of the World Health Organization classification of lymphoid neoplasms. Blood . 2016;127(20):2375-90.doi.org/10.1182/blood-2016-01-643569. Korgavkar K, Xiong M, Weinstock MA. Compact fluorescent lamps and risk of skin cancer. J Cutan Med Surg . 2013;17(5):308-12.doi.org/10.2310/7750.2013.12115. Seo AN, Lee SJ, Choi YH, Chung HY, Huh J, Yoon GS. Congenital primary cutaneous anaplastic large-cell lymphoma: a case report. Am J Dermatopathol . 2015;37(5):398-400.doi.org/10.1097/dad.0000000000000219. de Bruin PC, Beljaards RC, van Heerde P, Van Der Valk P, Noorduyn LA, Van Krieken JH, et al. Differences in clinical behaviour and immunophenotype between primary cutaneous and primary nodal anaplastic large cell lymphoma of T-cell or null cell phenotype. Histopathology . 1993;23(2):127-35.doi.org/10.1111/j.1365-2559.1993.tb00470.x. Yu JB, Blitzblau RC, Decker RH, Housman DM, Wilson LD. Analysis of primary CD30+ cutaneous lymphoproliferative disease and survival from the Surveillance, Epidemiology, and End Results database. J Clin Oncol . 2008;26(9):1483-8.doi.org/10.1200/jco.2007.14.1374. Woo DK, Jones CR, Vanoli-Storz MN, Kohler S, Reddy S, Advani R, et al. Prognostic factors in primary cutaneous anaplastic large cell lymphoma: characterization of clinical subset with worse outcome. Arch Dermatol . 2009;145(6):667-74.doi.org/10.1001/archdermatol.2009.74. Benner MF, Willemze R. Applicability and prognostic value of the new TNM classification system in 135 patients with primary cutaneous anaplastic large cell lymphoma. Arch Dermatol . 2009;145(12):1399-404.doi.org/10.1001/archdermatol.2009.280. Mesgarzadeh AH, Ghavimi MA, Gok G, Zarghami A. Infratemporal space infection following maxillary third molar extraction in an uncontrolled diabetic patient. J Dent Res Dent Clin Dent Prospects . 2012;6(3):113-5.doi.org/10.5681/joddd.2012.024. Brown RA, Fernandez-Pol S, Kim J. Primary cutaneous anaplastic large cell lymphoma. J Cutan Pathol . 2017;44(6):570-7.doi.org/10.1111/cup.12937. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 07 Mar, 2024 Reviews received at journal 26 Feb, 2024 Reviewers agreed at journal 24 Feb, 2024 Reviewers invited by journal 24 Feb, 2024 Editor assigned by journal 23 Feb, 2024 Editor invited by journal 22 Jan, 2024 Submission checks completed at journal 22 Jan, 2024 First submitted to journal 12 Jan, 2024 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-3855822","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":268559268,"identity":"13fc2743-fd74-4969-8aa2-7e69c60422c1","order_by":0,"name":"Xiaohan Yan","email":"","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":false,"prefix":"","firstName":"Xiaohan","middleName":"","lastName":"Yan","suffix":""},{"id":268559269,"identity":"7dbbea89-3fe7-4960-a07d-2b8e0de37a39","order_by":1,"name":"Wenhao Ren","email":"","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":false,"prefix":"","firstName":"Wenhao","middleName":"","lastName":"Ren","suffix":""},{"id":268559270,"identity":"1cfffd18-cd31-41ea-bd34-f5e5a1ff8ed7","order_by":2,"name":"Shaoming Li","email":"","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":false,"prefix":"","firstName":"Shaoming","middleName":"","lastName":"Li","suffix":""},{"id":268559271,"identity":"51e64d98-9647-463e-9bd0-f2ca20bb2b30","order_by":3,"name":"Zhuang Zhu","email":"","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":false,"prefix":"","firstName":"Zhuang","middleName":"","lastName":"Zhu","suffix":""},{"id":268559272,"identity":"e6e34450-6b48-49d7-9135-9820d1246b85","order_by":4,"name":"Ling Gao","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA8ElEQVRIie3RIWsDMRjG8acEMpM73eOg+QoJpwp1/SIJsxVTsw0cpGq+he07FArVNybOHNVxa81UJ0rNxIndXaka5DpXkT9vIIT3pwKEQvdY0RzVXh5IAXZ96SEDoyAAQtU/CDrCxG0kLl/K/b6uwafs5L4tRrFTg/OThyTVThttBeRHtBm/WWSJUyRdeohwM2m0aUgebdPIQq+dooT5yOdRGlW3hH21ZN5PHGsIFeCE0ZYo0UeSaiaX2mYQhGbj191QrqpDnvpIXFbi9FOPwBf5wR2fJzwuH9/PPnJtvi6AYTfdR90SN5f9UCgUCv3pF2NFS5qLnpbpAAAAAElFTkSuQmCC","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":true,"prefix":"","firstName":"Ling","middleName":"","lastName":"Gao","suffix":""},{"id":268559273,"identity":"93b78041-dffd-48fa-bd15-d9934721ed09","order_by":5,"name":"Keqian Zhi","email":"","orcid":"","institution":"the Affiliated Hospital of Qingdao University","correspondingAuthor":false,"prefix":"","firstName":"Keqian","middleName":"","lastName":"Zhi","suffix":""}],"badges":[],"createdAt":"2024-01-12 05:44:15","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-3855822/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-3855822/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":50120902,"identity":"9b8aa6fe-b5d7-4b44-87df-390da7893db9","added_by":"auto","created_at":"2024-01-24 19:38:17","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":33911,"visible":true,"origin":"","legend":"\u003cp\u003eAxial CT scan shows an extensive thickening in the subcutaneous tissue of the left temples.\u003c/p\u003e","description":"","filename":"1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-3855822/v1/2b607a1fff91e659d31ffd53.jpg"},{"id":50120904,"identity":"b383c337-0801-4fe7-bfd3-e0f18087ace7","added_by":"auto","created_at":"2024-01-24 19:38:17","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":242299,"visible":true,"origin":"","legend":"\u003cp\u003eHistopathology a sample of the left temples shows the tumor cells have an anaplastic morphology, with irregularly shaped nuclei, prominent nucleoli, and abundant cytoplasm (A-B). (Original magnification B 100 × , C 400×).\u003c/p\u003e","description":"","filename":"2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-3855822/v1/658db7a6c1d2fd416c37ea29.jpg"},{"id":50120905,"identity":"914a7c80-5bf2-4f6a-8a18-9e7ab1efbad1","added_by":"auto","created_at":"2024-01-24 19:38:17","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":1171052,"visible":true,"origin":"","legend":"\u003cp\u003eImmunohistochemical analysis showed positivity for (A) CD30 protein, (B) CD3 protein and (C) Ki67 protein. (Original magnification × 200). Immunohistochemical analysis showed negativity for (D) ALK(SP8) protein. (Original magnification × 200).\u003c/p\u003e","description":"","filename":"3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-3855822/v1/8fed9df6bb724e06cd2e11b0.jpg"},{"id":50121452,"identity":"c595faef-b29b-442e-91f6-bab222f01352","added_by":"auto","created_at":"2024-01-24 19:46:17","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":642691,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-3855822/v1/6661a0f0-b3a4-4464-9733-704d5250ec6c.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Primary cutaneous anaplastic large-cell lymphoma resembling space infection: a case report","fulltext":[{"header":"Background","content":"\u003cp\u003eAnaplastic large cell lymphoma (ALCL) is a rare type of non-Hodgkin\u0026apos;s lymphoma (NHL), as well as one of the subtypes of T cell lymphoma with ample cytoplasm and pleomorphic nuclei, expressing near-universal levels of the CD30[1, 2]. According to the 2016 WHO classification, there are four types of ALCL: systemic ALK-positive ALCL (ALK+ ALCL), systemic ALK-negative ALCL (ALK\u0026minus; ALCL), primary cutaneous ALCL (PC-ALCL), and breast implant-associated ALCL (BI-ALCL)[3].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e PC-ALCL presents on the skin and is characterized by a very good prognosis and response to treatment in the majority of cases. Early diagnosis of PC-ALCL is crucial because the clinical stage is directly related to the prognosis. Considering variations in morphology and immunophenotype, many differential diagnoses should be regarded as due to atypical lymphoma presentations. Here, we present a case of ALCL in a 78-year-old woman with temple and parotid gland region involvement, first thought to be a maxillofacial space infection. Therefore, we summarized some patient history experiences, specialty examination, clinical manifestations, and treatment, hoping to provide essential clues for diagnosing PC-ALCL with first symptoms in rare sites.\u0026nbsp;\u003c/p\u003e"},{"header":"Case presentation","content":"\u003cp\u003eA 78-year-old woman presented to\u003cs\u003e\u0026nbsp;\u003c/s\u003emaxillofacial surgery service with a 6-month history of swelling and pain in the left side of her face, which got worse in the past 20 days. 6 months ago, a biopsy was performed at another hospital, and the diagnosis was a chronic inflammatory. The patient had a 5-year history of diabetes mellitus.\u003c/p\u003e\n\u003cp\u003eClinical examination revealed diffused left-sided temporal swelling and unobvious\u0026nbsp;left parotid swelling, both with tough texture, obvious tenderness, and poor activity.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eLaboratory investigations found an elevated C-reactive protein (CRP) concentration of 109\u0026nbsp;\u0026middot;97 mg/dl\u0026nbsp;and a mildly raised procalcitonin concentration of 0.08 ng/ml.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eA cone beam computed tomography (CBCT) showed enlarged lymph\u0026nbsp;nodes of levels I to II in the left neck, extensive thickening of subcutaneous tissue of the left temples\u0026nbsp;[Fig.1], and a parotid hypodense lesion together with a blurred image of masticator, buccal, and submandibular space. Considering these findings, we suspected an infratemporal space infection. Based on these, an incision and drainage were planned for the patient. However, we found the lesion area with only a small amount of clear fluid rather than pus.\u0026nbsp;4 days later, the pain reappeared in\u0026nbsp;the lesion area.\u003c/p\u003e\n\u003cp\u003eThe definitive histopathological examination showed necrosis and extensive involvement of the left temples by a diffuse infiltrate containing large, atypical cells [Fig.2a, b]. Immunohistochemical stains showed that the cells were positive for CD30[Fig.3a], CD3[Fig.3b], Ki67[Fig.3c], Bcl-6, MUM1, c-Myc, p53, and EBER. They were negative for ALK (SP8) [Fig.3d], CD20, CD19, CD10, Bcl-2, CyclinD1, CD21, CD5, EBER(P), CD56, Pax-5, CD138, CD8 and CD4. To ensure no metastasis, a positron emission tomography (PET) scan was conducted, and no suspicious metastasis was found.\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;Considering the findings, the diagnosis of PC-ALCL was made. The patient was admitted to the lymphoma department for systemic chemotherapy with CDOP (cyclophosphamide, vindesine, liposome, dexamethasone, and prednisone), and the patient\u0026rsquo;s clinical status improved after the first treatment cycle.\u003c/p\u003e"},{"header":"Discussion and conclusions","content":"\u003cp\u003ePC-ALCL is a rare ALK-negative T-cell neoplasm with an annual incidence of 10 cases per million individuals[4]. It is commonly seen in people between 50 and 70, but it has also been reported in pediatric cases[5, 6]. Around 80% of cases present with a \u0026nbsp;localized cutaneous nodule or papule, and aggressive extracutaneous dissemination occurs in only about 10% of cases[7-9].\u0026nbsp;In this clinical case, the lesion occurring in the temple and parotid gland shows diffuse swelling with cumulative regional lymph nodes, combined with the history of diabetes mellitus and misdiagnosis of the first biopsy; all these factors associated with the clinical aspect of the lesion led us to the initially suspected of infratemporal space infection.\u003c/p\u003e\n\u003cp\u003eIn the diagnosis of infratemporal space infection, many diseases must be considered for differential diagnosis, such as temporomandibular joint disorder, lymphadenitis, and lymphoma[10]. We speculate that the patient\u0026rsquo;s pain relief temporarily after incision and drainage may be a decrease in local tension. In addition, clinical conditions do not resolve after the conventional treatment; other possibilities should be considered.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eNoteworthily, the diagnosis of ALCL requires complex interdisciplinary information integration by clinicians and pathologists[11]. Suppose no pus is found during the operation, we recommend carrying out a biopsy simultaneously, using HE staining, comprehensive IHC panels, and combining them with a systematic approach to diagnosing ALCL to prevent misdiagnosis.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003ePC-ALCL \u0026nbsp; Primary cutaneous anaplastic large-cell lymphoma\u003c/p\u003e\n\u003cp\u003eALCL\u0026nbsp;\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003eAnaplastic large cell lymphoma\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eCRP\u0026nbsp; C-reactive protein\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eCBCT\u0026nbsp; A cone beam computed tomography\u0026nbsp;\u003c/p\u003e\n\u003cp\u003ePET \u0026nbsp;positron emission tomography\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eAcknowledgements\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthor contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eXiaohan Yan performed drafting of the manuscript, writing, and literature search. Xiaohan Yan, Ling Gao, and Keqian Zhi contributed substantially to the conception, design, and patient data acquisition of the manuscript. Wenhao Ren and Shaoming Li performed the biopsy and was involved in revising the manuscript with respect to intellectual content. Zhuang Zhu performed the histopathological diagnosis and contributed to the interpretation of the data. All authors reviewed and approved the final manuscript prior to submission.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patient for publication of her clinical details and any accompanying images. A copy of the written consent is available on request. All of the data were anonymized.\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\u003eFinancial support\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study was funded by TaiShan Scholars Foundation of Shandong Province (No. tsqn202306397 [LG]).\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003ePletneva MA, Smith LB. Anaplastic large cell lymphoma: features presenting diagnostic challenges. \u003cem\u003eArch Pathol Lab Med\u003c/em\u003e. 2014;138(10):1290-4.doi.org/10.5858/arpa.2014-0295-CC.\u003c/li\u003e\n\u003cli\u003eZhang XR, Chien PN, Nam SY, Heo CY. Anaplastic Large Cell Lymphoma: Molecular Pathogenesis and Treatment. \u003cem\u003eCancers (Basel)\u003c/em\u003e. 2022;14(7).doi.org/10.3390/cancers14071650.\u003c/li\u003e\n\u003cli\u003eSwerdlow SH, Campo E, Pileri SA, Harris NL, Stein H, Siebert R, et al. The 2016 revision of the World Health Organization classification of lymphoid neoplasms. \u003cem\u003eBlood\u003c/em\u003e. 2016;127(20):2375-90.doi.org/10.1182/blood-2016-01-643569.\u003c/li\u003e\n\u003cli\u003eKorgavkar K, Xiong M, Weinstock MA. Compact fluorescent lamps and risk of skin cancer. \u003cem\u003eJ Cutan Med Surg\u003c/em\u003e. 2013;17(5):308-12.doi.org/10.2310/7750.2013.12115.\u003c/li\u003e\n\u003cli\u003eSeo AN, Lee SJ, Choi YH, Chung HY, Huh J, Yoon GS. Congenital primary cutaneous anaplastic large-cell lymphoma: a case report. \u003cem\u003eAm J Dermatopathol\u003c/em\u003e. 2015;37(5):398-400.doi.org/10.1097/dad.0000000000000219.\u003c/li\u003e\n\u003cli\u003ede Bruin PC, Beljaards RC, van Heerde P, Van Der Valk P, Noorduyn LA, Van Krieken JH, et al. Differences in clinical behaviour and immunophenotype between primary cutaneous and primary nodal anaplastic large cell lymphoma of T-cell or null cell phenotype. \u003cem\u003eHistopathology\u003c/em\u003e. 1993;23(2):127-35.doi.org/10.1111/j.1365-2559.1993.tb00470.x.\u003c/li\u003e\n\u003cli\u003eYu JB, Blitzblau RC, Decker RH, Housman DM, Wilson LD. Analysis of primary CD30+ cutaneous lymphoproliferative disease and survival from the Surveillance, Epidemiology, and End Results database. \u003cem\u003eJ Clin Oncol\u003c/em\u003e. 2008;26(9):1483-8.doi.org/10.1200/jco.2007.14.1374.\u003c/li\u003e\n\u003cli\u003eWoo DK, Jones CR, Vanoli-Storz MN, Kohler S, Reddy S, Advani R, et al. Prognostic factors in primary cutaneous anaplastic large cell lymphoma: characterization of clinical subset with worse outcome. \u003cem\u003eArch Dermatol\u003c/em\u003e. 2009;145(6):667-74.doi.org/10.1001/archdermatol.2009.74.\u003c/li\u003e\n\u003cli\u003eBenner MF, Willemze R. Applicability and prognostic value of the new TNM classification system in 135 patients with primary cutaneous anaplastic large cell lymphoma. \u003cem\u003eArch Dermatol\u003c/em\u003e. 2009;145(12):1399-404.doi.org/10.1001/archdermatol.2009.280.\u003c/li\u003e\n\u003cli\u003eMesgarzadeh AH, Ghavimi MA, Gok G, Zarghami A. Infratemporal space infection following maxillary third molar extraction in an uncontrolled diabetic patient. \u003cem\u003eJ Dent Res Dent Clin Dent Prospects\u003c/em\u003e. 2012;6(3):113-5.doi.org/10.5681/joddd.2012.024.\u003c/li\u003e\n\u003cli\u003eBrown RA, Fernandez-Pol S, Kim J. Primary cutaneous anaplastic large cell lymphoma. \u003cem\u003eJ Cutan Pathol\u003c/em\u003e. 2017;44(6):570-7.doi.org/10.1111/cup.12937.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-oral-health","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ohea","sideBox":"Learn more about [BMC Oral Health](http://bmcoralhealth.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ohea/default.aspx","title":"BMC Oral Health","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Primary cutaneous anaplastic large-cell lymphoma, space infection, temporal and parotid gland region, case report","lastPublishedDoi":"10.21203/rs.3.rs-3855822/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-3855822/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground: \u003c/strong\u003ePrimary cutaneous anaplastic large-cell lymphoma (PC-ALCL) is a rare T-cell lymphoma belonging to the CD30+ T-cell lymphoproliferative disorders. \u0026nbsp;The case of PC-ALCL in the temporal and parotid gland region is exceedingly rare. Herein, we report a case of PC-ALCL involving the temporal and parotid gland region mimicking maxillofacial space infection.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase presentation: \u003c/strong\u003eA 78-year-old woman presented to\u003cdel\u003e \u003c/del\u003e\u0026nbsp;maxillofacial surgery service with a 6-month history of swelling and pain in the left side of her face. The patient underwent incision and drainage, and we unexpectedly found no pus in the lesion area. Incisional biopsy showed necrosis and extensive involvement of the left temples by a diffuse infiltrate containing large, atypical cells. \u0026nbsp;The tumor cells were positive for CD30, CD3, and Ki67, and negative for ALK (SP8). \u0026nbsp;After considering these findings, a diagnosis of PC-ALCL was rendered. The patient was admitted to the lymphoma department for systemic chemotherapy and no relapse occurred during a follow-up period of three months.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusions: \u003c/strong\u003eThis report suggests that if there are suspicious intraoperative manifestations, carrying out a biopsy simultaneously, using HE staining, comprehensive IHC panels, and combining them with a systematic approach is essential to diagnosing ALCL to prevent misdiagnosis.\u003c/p\u003e","manuscriptTitle":"Primary cutaneous anaplastic large-cell lymphoma resembling space infection: a case report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-01-24 19:38:12","doi":"10.21203/rs.3.rs-3855822/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2024-03-07T07:21:11+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-02-26T08:29:48+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"cc016efd-0cdd-439c-bb1b-745966ea6bad","date":"2024-02-24T20:01:16+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-02-24T09:12:57+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-02-23T15:19:20+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2024-01-22T13:34:49+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2024-01-22T13:33:49+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Oral Health","date":"2024-01-12T05:37:34+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-oral-health","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ohea","sideBox":"Learn more about [BMC Oral Health](http://bmcoralhealth.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ohea/default.aspx","title":"BMC Oral Health","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"9a116ad9-ab82-4133-a31a-dfd722129889","owner":[],"postedDate":"January 24th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2024-03-25T05:59:37+00:00","versionOfRecord":[],"versionCreatedAt":"2024-01-24 19:38:12","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-3855822","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-3855822","identity":"rs-3855822","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

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