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Castleman’s disease, Intra Parotid, Salivary gland, IntroductionCastleman&,rsquo s disease (CD) is a rare non&,#8208 neoplastic lymphoproliferative disorder, first known in 1956 by Dr. Benjamin Castleman who described a 40-year-old male with a mediastinal lymph node mass characterized histologically by lymph node hyperplasia and follicles with hyalinized centers [ 1, ]. This disease can occur in any lymphatic chain, and mediastinum is the most common site (60-86%). Salivary gland involvement is greatly rare (6-14%) and very few cases have been reported to date [ 2, ]. Because of unknown etiology, it is represented by various terminologies such as lymphoid hamartoma, giant lymph node hyperplasia, angiofollicular hamartoma, and benign giant lymphoma [ 3, ].The CD diagnosis is basically confirmed by biopsy. The characteristic histopathological criteria included angiofollicular lymph node hyperplasia in a lymph node region [ 4, ]. Due to the infrequency of CD, the data obtained from literature is more from case series and retrospective studies. In this report, we describe a rare case of unicentric CD presenting in left parotid gland as the first manifestation. Due to an increase risk of the development of lymphoma, complete diagnostic workup including clinical presentation, radiological imaging, and histopathological assessment should be performed very early to arrive at an accurate diagnosis and to provide appropriate therapy.Materials and MethodA 15-year-old boy patient referred to Plastic Surgery Department at Imam Khomeini Cancer Institute with chief complaint of left facial swelling for past 4 years (Figure 1a, and b,). He had history of trauma for 5days. On physical examination, the mass was 1&,times 1cm oval and bean shaped, firm, non-tender with no sign of inflammation, which was snaky in onset and progressed slowly. Paranasal bones encroachment were unremarkable. The parotid duct in the affected side had normal physical appearance without debris or purulent discharge. In the first visit, diagnosis of mucocele was made and aspiration was carried out but it was negative. After one week of follow-up, no changes were observed in mass size and further clinicoradiological evaluation was administered. In the axial and coronal computed tomography (CT) scan slices without contrast, multiple small adenopathy were seen at the both side of neck. There was also a small well-defined hypo dense solid lesion adjacent to the left masseter muscle and in the superficial lobe of left parotid gland. No calcification or necrotic areas were seen within the lesion (Figure 2,).Figure 1.Clinical aspect of the patient showing slight expansion in left parotid gland. a, Frontal, b, Oblique viewFigure 2. Computed tomography without contrast (coronal view) showing small well-defined hypo dense solid lesion adjacent to the left masseter muscle and in the superficial lobe of left parotid glandFollowing the CTs, ultrasonography (US) examination was administrated for the patient and in the US findings an oval and bean-shaped mass measuring approximately 30*6.5 mm accompanied by centripedal vascularity in the buccal space was reported. In addition, presence of multiple giant lymph nodes with increased wall thickness in the both sides of jugular space and submandibular space was noted. The patient underwent a left superficial parotidectomy with preservation of the facial nerve under general anesthesia. On gross examination tangray salivary gland was identified, which showed central area of well-demarcated gray nodule with firm consistency. Microscopic examination of referred specimen revealed salivary gland tissue composed of mostly serous acini and related ducts with preserved architecture and intra parotid lymph node with marked vascular proliferation and hyalinization of germinal center with appearance corresponded to transform germinal centers regressively. Concentric aggregation of lymphocytes at the mantle zone was noted. Interfollicular stroma showed multiple vessels admixed with few plasma cells. Immuno histo chemistry (IHC) study for CD-21 showed strong positive in follicular dendritic cells within germinal center, which confirmed diagnosis. Postoperatively, the patient did well, and he experienced no major complications, that the authors obtained written and signed informed consent&,nbsp from patient&apos,s guardians for publishing this&,nbsp case report.DiscussionCD is a rare pathological disorder that can rarely affect the parotid gland [ 5, ].Based on analysis of American databases, the incidence of CD is estimated to be 21-25 cases per million person-year [ 6, ]. The most common location of CD is the thorax followed by the neck and abdomen. Within the head and neck region, CD usually presents either as a single nodule under the sternocleidomastoid muscle or as a mass affecting mediastinum to cervical area. Involvement of the salivary gland is considerably infrequent and even more rarely in the parotid gland [ 7, ]. In our literature review, based on available information, parotid was the most common site of involvement compared to other salivary glands. In the present case, the lesion was located in buccal side of left parotid gland mimicking a salivary gland tumor. Several theories have been proposed for the un known etiology of this disease. The strongest hypotheses indicate robust relation between virus or chronic inflammation and lymphoproliferation [ 3, ]. In 1970, Flendrig et al. [ 8, ] identified distinct histopathologic variants of this lesion and two years later, Keller et al. [ 9, ] classified it into the hyaline vascular (HV) and plasma cell types some patients have a mixed variant. Clinically, CD is sub classified into unicentric (UCD) and multicentric (MCD) types. The UCD form usually presents as an asymptomatic painless palpable enlarged lymph node mass which reflects its benign feature. On the other hand, MCD is more aggressive and presents with systemic symptoms such as fever, loss of weight, and splenomegaly and can be associated with some malignancies, such as Kaposi&,rsquo s sarcoma, non-Hodgkin&,rsquo s lymphoma, Hodgkin&,rsquo s disease, and POEMS syndrome (polyneuropathy, organomegaly, endocrinopathy, M protein and skin changes) [ 3, ]. In our review, we found 57 cases most of them had an asymptomatic swelling similar to the presented case. However, three patients showed symptoms and only one of them suffered from intermittent jaundice.According to histopathological pattern, the HV is the most common subtype, accounting for 80-90 % of the cases [ 2, ]. This variant occurs over broad age range and, in most studies, males and females were equally affected. HV-CD can be divided into two different groups, follicular and interfollicular region changes. In classical HV-CD, the overall lymph node is preserved but inflected. Lymphoid follicles grow, scatter throughout the cortex and medulla, and commonly consist of two or more small germinal centers (socalled &,ldquo twinning&,rdquo ) [ 10, ]. In our case, sclerotic blood vessels radially penetrated the germinal centers, which are known as &,ldquo lollipop lesions&,rdquo (Figure 3a, and b,). Twenty two percent of the localized and plurality of MCD is plasma cell type. The mixed type is scarce and characterized by a mixture of the two types [ 2, ]. Of the 58 specimens, 54 were HV (93.1%), two were mixed (3.4%) and two were plasma-cell type (3.4%). Regarding to clinicopathologic correlation, the main differential diagnosis were included toxoplasma lymphadenitis, reactive lymphadenitis and for more severe lesions, follicular lymphoma and mantle cell lymphomas were considered. The formers are excluded by means of large sized follicles, well-formed light and dark zones, tangible body macrophages, and intact sinuses while the latter ones are ruled out by presence monotonous population of neoplastic cell with lack of prominent vascularity in interfollicular region [ 10, ]. A review literature of available published cases for CD revealed more than 50 cases their features are summarized in Table 1,. This information reflects the rare incidence of called disease. Since the disease shows nonspecific clinical, radiological, or cytological features the diagnosis is challenging and should be made by the aid of histopathological pattern as it was mentioned earlier. Radiographic findings can be helpful for differential diagnosis. CTs with contrast showed a dense enhanced oval, homogenous mass, which reflect hyper vascularity of these lesions. US is also feasible to confirm these features. The prognosis of CD is unpredictable and depends fundamentally on disease subtype [ 11, ].NameYearSexAgeLocationu/m centricHistology patternClinical courseTreatmentCavallaro, et al. [ 13, ]1985F23ParotidUHVAsymptomaticComplete excisionPrasad H, et al. [ 14, ]1988M60ParotidUPlasma cellTendernessComplete excisionWoolgar, et al. [ 15, ]1991F24ParotidUHVAsymptomaticSuperficial parotidectomyLatz, et al. [ 16, ]1992M38ParotidUHVAsymptomaticRadiationYi, et al. [ 17, ]1995M33ParotidUHVAsymptomaticComplete excisionAhuja, et al. [ 18, ]1995M50ParotidUHVAsymptomaticComplete excision, lymphadenectomyYoo, et al. [ 19, ]1995M8SMGUHVAsymptomaticComplete excisionLeocata, et al. [ 20, ]1996F16ParotidUHVAsymptomaticComplete excisionChoi, et al. [ 21, ]1997M14ParotidUHVAsymptomaticSuperficial parotidectomyGoodisson, et al. [ 11, ]1997M36ParotidUHVAsymptomaticSuperficial parotidectomySantonja, et al. [ 22, ]1997F5ParotidUHVAsymptomaticComplete excisionPanayiotides, et.al. [ 23, ]1998M30ParotidUHVAsymptomaticComplete excisionParez, et al. [ 24, ]1999M9SMGUHVAsymptomaticComplete excisionNahlieli, et al. [ 25, ]2000F18ParotidUHVAsymptomaticComplete excisionSanchez-Cuellar, et al. [ 26, ]2001M19ParotidUHVAsymptomaticSuperficial parotidectomyMohan, et al. [ 27, ]2003M17ParotidUHVAsymptomaticSuperficial parotidectomySamadi et al. [ 28, ]2003M6ParotidUHVAsymptomaticComplete excisionKilty, et al. [ 29, ]2005F22ParotidUHVAsymptomaticSuperficial parotidectomyM26UHVTendernessSuperficial parotidectomyF50MPlasma cellAsymptomaticSuperficial parotidectomyDursun, et al. [ 30, ]2006M45ParotidUHVAsymptomaticComplete excisionAkdogan, et al. [ 31, ]2006F52Bilateral parotidUHVAsymptomaticComplete excisionPark, et al. [ 32, ]2008M9ParotidUHVAsymptomaticSuperficial parotidectomyErdogan, et al. [ 33, ]2008F15ParotidUHVAsymptomaticComplete excisionLee, et al. [ 34, ]2009F29ParotidUHVAsymptomaticSuperficial parotidectomyGurbuzler, et al. [ 3, ]2010F34ParotidUHVAsymptomaticParotidectomyZhong, et al. [ 35, ]20104M/6F10 patients between 13-54Parotid and neck region.UHVAsymptomaticComplete excisionLin, et al. [ 36, ]2010F18ParotidUHVAsymptomaticComplete excisionErkan, et al. [ 37, ]2011F29ParotidUHVAsymptomaticSuperficial parotidectomyMayada&,#287 l&,#305 , et al. [ 38, ]2012M45ParotidUHVMalaiseSubtotal parotidectomy with radiotherapyReece, et al. [ 5, ]2012M46ParotidUHVAsymptomaticComplete excision&,Uuml st&,uuml n, et al. [ 39, ]2012F52ParotidUHVAsymptomaticSuperficial parotidectomyTemirbekov, et al.[ 40, ]2013F35ParotidUHVAsymptomaticSuperficial parotidectomyKumar. et al [ 41, ]2014M15ParotidUHVAsymptomaticComplete excisionBollig, et al. [ 42, ]2014M14ParotidUHVAsymptomaticSuperficial parotidectomyIaconetta, et al. [ 12, ]2014F35ParotidUHVAsymptomaticSuperficial parotidectomyDelaney, et al. [ 7, ]2015M7ParotidUHVAsymptomaticSuperficial parotidectomyF11ParotidUHVAsymptomaticSuperficial parotidectomyKishori, et al. [ 43, ]2015M47SMGUHVasymptomaticComplete excisionAbo-Alhassan, et al. [ 44, ]2015F29ParotidUHVAsymptomaticComplete excisionShah, et al. [ 45, ]2015F22SMGUHVAsymptomaticComplete excisionMalzone, et al. [ 46, ]2016M44SMGUHVAsymptomaticComplete excisionLin, et al.[ 47, ]2016F46ParotidUHVAsymptomaticParotidectomyHamilton, et al.[ 48, ]2017F12SMGUHVAsymptomaticComplete excisionEkmekci, et al. [ 6, ]2018M59ParotidMmixedAsymptomaticSuperficial parotidectomyZhai, et al. [ 49, ]2019F62ParotidUmixedIntermittent jaundiceComplete excisionBatra, et al. [ 2, ]2019M35ParotidUHVAsymptomaticSuperficial parotidectomyXiao-Dong, et al. [ 50, ]2020M39ParotidUHVAsymptomaticSuperficial parotidectomyF, female, SMG, submandibular Gland, U, unicentric, HV, hyaline vascular |