pissn 2384-1095 eissn 2384-1109 imri 2018;22:240-244 A Rre Mnifesttion of Solitry Primry Bone Lymphom of the Finger: Cse Report Jeong A Yeom 2, You Seon Song 1 *, In Sook Lee 1, Kyung Un Choi 3, Jeung Il Kim 4 1 Deprtment of Rdiology, Pusn Ntionl University Hospitl, Busn, Kore 2 Deprtment of Rdiology, Pusn Ntionl University Yngsn Hospitl, Busn, Kore 3 Deprtment of Pthology, Pusn Ntionl University Hospitl, Busn, Kore 4 Deprtment of Orthopedic Surgery, Pusn Ntionl University Hospitl, Busn, Kore Cse Report Received: Novemer 19, 2018 Revised: Decemer 11, 2018 Accepted: Decemer 12, 2018 Correspondence to: You Seon Song, M.D. Deprtment of Rdiology, Pusn Ntionl University Hospitl, 179 Gudeok-ro, Seo-gu, Busn 49241, Kore. Tel. +82-51-240-7354 Fx. +82-51-244-7534 E-mil: yssongrd@gmil.com This is n Open Access rticle distriuted under the terms of the Cretive Commons Attriution Non-Commercil License (http://cretivecommons.org/licenses/ y-nc/3.0/) which permits unrestricted non-commercil use, distriution, nd reproduction in ny medium, provided the originl work is properly cited. Copyright 2018 Koren Society of Mgnetic Resonnce in Medicine (KSMRM) Primry extrnodl one lymphom involving the peripherl extremities is extremely rre. Here, we report definitive cse of diffuse lrge B-cell lymphom involving the phlngel one of the 3rd finger. Systemic evlution reveled the lesion s the only site of lymphom involvement. Keywords: Primry lymphom; Bone; Finger INTRODUCTION Primry one lymphom is rre disese, ccounting for less thn 5% of primry one tumors nd 3% of mlignnt one tumors (1, 2). Most of the primry one lymphoms represent non-hodgkin lymphom, especilly diffuse lrge B-cell lymphom (DLBCL) (3). Cliniclly, one lymphoms present s plple msses with intermittent one pin (4). According to previous study (5), the metdiphysis of the femur is the most common site involved. Other sites include pelvis, tii, hed nd neck, nd verter. Lymphom loclized to peripherl sites such s the feet nd the hnds, or the toes nd the fingers, is rrely reported. Here, we report very rre cse of solitry primry one lymphom locted in the middle phlnx of the third finger. CASE REPORT A 71-yer-old mle presented to our institution complining of swelling in the right third finger, with six-month history of symptoms. He hd no specific medicl history. Lortory testing reveled elevted erythrocyte sedimenttion rte nd C-rective protein (51 mm/h nd 5.8 mg/dl, respectively). Plin rdiogrph reveled evidence of ony mss cusing corticl disruption t the rdil spect of the third middle phlnx (Fig. 1). The ptient underwent mgnetic resonnce imging (MRI) for further evlution. T2-weighted MRI showed well-defined mss of high signl intensity, involving the djcent 3rd middle phlngel one with lrge soft tissue component. The mss ws thought to hve originted in the one rther thn soft tissue, ecuse the center of the mss ws ner the cortex of the phlngel one nd norml corticl 240
one ws noted within the oundry of the mss. The mss utted with the flexor tendon of the third finger (Fig. 1). T1-weighted imging reveled mss of intermedite signl intensity (Fig. 1c). The contrst-enhnced MRI showed strong nd homogeneous enhncement (Fig. 1d). Bsed on the imging findings, the differentil dignosis included mimics of primry one tumor such s glomus tumor or introsseous epiderml inclusion cyst nd metsttic one tumor, despite its rrity. Totl excision ws performed for symptom relief. c d Fig. 1. A 71-yer-old-mn with DLBCL involving 3rd finger. () Plin rdiogrph shows osteolytic ony mss (rrows) with soft-tissue component (rrowheds) involving right third middle phlnx. () Axil T2-weighted imge revels highsignl intensity mss (rrows) ssocited with the right third middle phlnx. The mss originted in the intrmedullry cvity constituting the lrge soft-tissue component, utting the flexor tendon of the third finger. (c) Coronl T1-weighted imge indictes mss of intermedite signl intensity (rrow). (d) Coronl T1-weighted imge with ft suppression fter gdolinium dministrtion shows homogenous enhncement of the mss (rrow). 241
Primry Bone Lymphom of the Finger Jeong A Yeom, et l. A yellowish-white soft tissue mss cusing lysis of the middle phlngel one ws visile in the opertive field. Stining with Hemtoxylin nd Eosin (H&E) reveled diffuse infiltrtion of typicl cells (Fig. 2). Immunohistochemicl nlysis showed positive test results for leukocyte common ntigen (LCA) (Fig. 2) nd CD20, nd negtive findings for CD45R0, pn cytokertin, synptophysin, CD56, nd CD99. Therefore, the finl pthologic dignosis ws DLBCL. Chest nd dominl computed tomogrphy (CT) scn, one scn (Fig. 3), nd positron emission tomogrphy (PET)-CT (Fig. 3) were performed to determine the involvement of other orgns. However, no evidence of lymphom involvement ws detected t ny other site, nor ws ny evidence of disese spred to other orgns detected during the 6 months of follow-up. Fig. 2. Microscopic findings of the finger mss. () Microscopic finding (Hemtoxylin & Eosin stin, 200) shows diffuse infiltrtion of typicl cells. () Immunohistochemicl stining ws positive for leukocyte common ntigen. Fig. 3. Systemic evlution of other orgn involvement. () Bone scn revels no evidence of other one involvement. () PET-CT revels no evidence of other orgn involvement of lymphom. 242
DISCUSSION Lymphom is the most common hemtologic disorder of the lymphoreticulr tissue. However, lymphoms cn lso involve orgns outside of the lymphtic system, referred to s extrnodl lymphom. Extrnodl lymphom my occur in the gonds, gstrointestinl trct, lung, centrl nervous system nd one. In primry one lymphom, xil rther thn ppendiculr skeleton is more commonly ffected (6). Moreover, the smll ones of the peripherl extremities re very rrely involved in one lymphom. To our knowledge, only few cses (7), which were confirmed s primry one lymphom, involved the hnd. The dignostic criteri include the presence of lymphom involving n osseous site, without evidence of ny other orgn involvement for t lest 6 months fter the initil dignosis (8). The regionl lymph node involvement cnnot exclude dignosis of primry one lymphom. In our cse, the plple mss on the third finger ws the initil mnifesttion of the disese, without ny msses involving other orgns on systemic evlution. The ptient visited our hospitl in My, following initil plin rdiogrph t nother hospitl in Jnury of the sme yer, showing the finger mss. Therefore, no evidence of metsttic spred ws found out 6 months fter the initil presenttion. Thus, it is resonle to define our cse s primry one lymphom. According to previous report (6), the mjority of primry one lymphom occurs in older dults, nd more thn 90% of ptients re over the ge of 30 yers. In lrge series, ptients dignosed with primry one DLBCL were younger (medin ge, 56 yers) thn ptients with nonosseous DLBCL nd secondry one lymphom (9). In our cse, the ptient ws 71 yers old. Additionlly, men re more frequently ffected thn women, with rtio rnging from 1.2 to 1.8 (6). Femur is the most common site of involvement in nerly 50% of the cses. The pelvis is ffected in out 20%, nd other sites including the verter, ris nd tii ccount for the reminder (5). DLBCL is the most common histologicl sutype of primry one lymphom, s in our cse (6). Rrely, folliculr lymphom, smll lymphocytic lymphom nd mrginl zone lymphom my mnifest s primry one lymphom. Imging findings of primry one lymphom vry, nd my e non-specific in some ptients (4). Although, the imging findings of one lymphom exhiit wide spectrum, few fetures re chrcteristic of the disese. Typiclly, lymphomtous one involvement is defined y solitry lytic lesion ner the end of long one with permetive or moth-eten ppernce, nd ggressive periostel rection (10). In ddition, extensive surrounding soft-tissue msses without significnt corticl destruction hs een reported exclusively in round cell tumors including one lymphom, multiple myelom nd Ewing srcom (3). However, in our cse, none of these typicl imging findings of one lymphom ws detected. Therefore, the initil rdiologic dignosis ws not focused on lymphom, ut rther on the more commonly encountered msses ssocited with the finger. Here, we reported very rre cse of solitry primry one lymphom involving the third finger. Although rre with unusul loction nd imging findings, the possiility of this disese should e considered to void misdignosis. REFERENCES 1. Krishnn A, Shirkhod A, Tehrnzdeh J, Armin AR, Irwin R, Les K. Primry one lymphom: rdiogrphic-mr imging correltion. Rdiogrphics 2003;23:1371-1383; discussion 1384-1387 2. Poirci O, Rosc E, Poirci DD, Nichit A. Primry diffuse lrge B-cell lymphom of the humerus. Cse report nd review of literture. Med Con 2014;9:69-72 3. Mullign ME, McRe GA, Murphey MD. Imging fetures of primry lymphom of one. AJR Am J Roentgenol 1999;173:1691-1697 4. de Cmrgo OP, dos Sntos Mchdo TM, Croci AT, et l. Primry one lymphom in 24 ptients treted etween 1955 nd 1999. Clin Orthop Relt Res 2002:271-280 5. Vincent JM, Ng YY, Norton AJ, Armstrong P. Cse report: primry lymphom of one--mri ppernces with pthologicl correltion. Clin Rdiol 1992;45:407-409 6. Jwd MU, Schneideruer MM, Min ES, Cheung MC, Koniris LG, Scully SP. Primry lymphom of one in dult ptients. Cncer 2010;116:871-879 7. Glti V, Wortmnn F, Stng FH, Thorns C, Milnder P, Kisch T. A rre mnifesttion of primry one lymphom: solitry diffuse lrge B-cell lymphom of the little finger. J Hnd Surg Am 2018;43:779 e771-779 e774 8. Coley BL, Higinothm NL, Groeseck HP. Primry reticulum-cell srcom of one; summry of 37 cses. Rdiology 1950;55:641-658 9. Li X, Xu-Monette ZY, Yi S, et l. Primry one lymphom exhiits fvorle prognosis nd distinct gene expression 243
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