scholarly journals Castleman’s disease presenting as parotid mass: a case report

Author(s):  
Ankur Batra ◽  
Megha Goyal

<p class="abstract">Castleman’s disease is a rare, benign, lymphoproliferative disorder of unknown etiology. It can involve any lymph node group in the body with mediastinum being the commonest site. Salivary glands are affected rarely. We report a<strong> </strong>35 year old male patient who presented with slowly progressive, painless right sided parotid swelling for 3 years. Clinical examination showed a 4×3 cm single, firm, non-tender, non-pulsatile swelling with smooth surface and normal overlying skin present in the right parotid region. Magnetic resonance imaging (MRI) revealed a well-defined hyper-intense mass lesion on T2 measuring 3.9×3×2.8 cm and Fine needle aspiration cytology (FNAC) showed intense lymphoplasmacytic infiltrates. No conclusive diagnosis could be made on the basis of FNAC. So, the excision of parotid mass with facial nerve preservation was done. The final histopathology confirmed the diagnosis as Castleman’s disease.<strong> </strong>Although Castleman’s disease in the parotid gland is rare, clinicians should consider it as the differential diagnosis of any solid tumors that exhibit non-specific presenting characteristics and surgical excision is preferred treatment for unicentric disease. <strong></strong></p>

2009 ◽  
Vol 9 ◽  
pp. 940-945 ◽  
Author(s):  
Hajer Racil ◽  
Sana Cheikh Rouhou ◽  
Olfa Ismail ◽  
Saoussen Hantous-Zannad ◽  
Nawel Chaouch ◽  
...  

Castleman's disease (CD) is an uncommon, mainly benign, lymphoproliferative disorder of unknown etiology, mostly involving the mediastinum. Parenchymal lung involvement of the disease is exceedingly rare. We describe a case of CD in a 23-year-old woman with a 4-year history of recurring dyspnea and nonproductive cough, whose chest X-ray showed an abnormal shadow of the right hilum. Chest computed tomography confirmed the presence of a tissue-density mass of the right lower lobe, demonstrating poor contrast enhancement, associated with multiple laterotracheal and mediastinal lymphadenopathies. The patient underwent curative surgery, revealing a right hilar compressive mass, with an intrafissural development between the superior and middle lobes. Pneumonectomy was performed due to profuse bleeding. This case of CD is particular because of its unusual intrapulmonary location and its intrafissural development. Poor contrast enhancement is atypical in CD.


2016 ◽  
Vol 2016 ◽  
pp. 1-3 ◽  
Author(s):  
Rafael Parra-Medina ◽  
José Ismael Guio ◽  
Patricia López-Correa

Castleman’s disease (CD) is a rare lymphoproliferative disorder of unknown etiology. It typically occurs in adulthood but it may also develop in childhood. Clinically, this disease may be classified as localized (unicentric) or systemic (multicentric). Six cases of breast CD have been described in the literature, and all have been reported in adults. Herein we describe the case of a 15-year-old female who presented with a slow-growing tumor in the right breast. The tumor was excised and histopathological examination demonstrated hyaline vascular variant CD. After two years of follow-up, the patient was asymptomatic without evidence of cervical or axillary lymphadenopathy.


2015 ◽  
Vol 2015 ◽  
pp. 1-4 ◽  
Author(s):  
Sean W. Delaney ◽  
Shengmei Zhou ◽  
Dennis Maceri

Introduction. Angiofollicular lymph node hyperplasia (Castleman’s disease) is a nonmalignant lymphoproliferative disorder that generally involves the lymph nodes of young adults, most commonly in the mediastinum. Rarely, Castleman’s disease may present in the parotid gland. The disease can be further classified into unicentric or multicentric forms, with considerable differences in presentation, treatment, and prognosis.Case(s). We present cases of two pediatric patients, aged 7 and 11, who both presented with a slow-growing, painless parotid mass. In each case, the mass was excised via a superficial parotidectomy and the diagnosis made postoperatively upon further pathologic examination. At 6 months of follow-up, both had fully intact facial nerve function and no evidence of recurrence.Discussion. Castleman’s disease presents a diagnostic challenge in the head and neck region, as radiographic characteristics and fine needle aspiration results are often inconclusive. Definitive diagnosis requires surgical excision for pathologic examination. The unicentric form generally presents as a painless mass and can be successfully treated with complete excision. The multicentric form is associated with constitutional symptoms and its treatment remains controversial.Conclusion. Although rare, clinicians should be aware of both forms of Castleman’s disease when creating a differential diagnosis for parotid masses.


2021 ◽  
Vol 22 (1) ◽  
Author(s):  
Ryosuke Saiki ◽  
Kan Katayama ◽  
Yosuke Hirabayashi ◽  
Keiko Oda ◽  
Mika Fujimoto ◽  
...  

Abstract Background Multicentric Castleman’s disease is a life-threatening disorder involving a systemic inflammatory response and multiple organ failure caused by the overproduction of interleukin-6. Although renal complications of Castleman’s disease include AA amyloidosis, thrombotic microangiopathy, and membranoproliferative glomerulonephritis, membranous nephropathy is relatively rare. We experienced a case of secondary membranous nephropathy associated with Castleman’s disease. Case presentation The patient was a 43-year-old Japanese man who had shown a high zinc sulfate value in turbidity test, polyclonal hypergammaglobulinemia, anemia, and proteinuria. A physical examination revealed diffuse lymphadenopathy, an enlarged spleen and papulae of the body trunk. A skin biopsy of a papule on the patient’s back showed plasma cells in the perivascular area and he was diagnosed with multicentric Castleman’s disease, plasma cell variant. Kidney biopsy showed the appearance of bubbling in the glomerular basement membranes in Periodic acid methenamine silver stain and electron microscopy revealed electron dense deposits within and outside the glomerular basement membranes. Since immunofluorescence study showed predominant granular deposition of IgG1 and IgG2, he was diagnosed with secondary membranous nephropathy associated with Castleman’s disease. He was initially treated with prednisolone alone, however his biochemical abnormalities did not improve. After intravenous tocilizumab (700 mg every 2 weeks) was started, his C-reactive protein elevation, anemia, and polyclonal gammopathy improved. Furthermore, his urinary protein level declined from 1.58 g/gCr to 0.13 g/gCr. The prednisolone dose was gradually tapered, then discontinued. He has been stable without a recurrence of proteinuria for more than 6 months. Conclusions Tocilizumab might be a treatment option for secondary membranous nephropathy associated with Castleman’s disease.


2013 ◽  
Vol 2013 ◽  
pp. 1-5 ◽  
Author(s):  
Hideki Ota ◽  
Hideki Kawai ◽  
Tsubasa Matsuo

Castleman's disease is an uncommon lymphoproliferative disorder of unknown etiology, most often involving the mediastinum. It has 2 distinct clinical forms: unicentric and multicentric. Unicentric Castleman's disease arising from an intrapulmonary lymph node is rare, and establishing a preoperative diagnosis of this disease is very difficult mainly due to a lack of specific imaging features. We report a case of intrapulmonary unicentric Castleman's disease in an asymptomatic 19-year-old male patient who was accurately diagnosed by preoperative computed tomography (CT). The mass was incidentally found on a routine chest X-ray. A subsequent dynamic CT showed a well-defined, hypervascular, soft-tissue mass with small calcifications located in the perihilar area of the right lower lung. Three-dimensional CT (3D-CT) angiography indicated that the mass was receiving its blood supply through a vascular network at its surface that originated from 2 right bronchial arteries. The clinical history and CT findings were consistent with a diagnosis of unicentric Castleman's disease, and we safely and successfully removed the tumor via video-assisted thoracoscopic surgical lobectomy. This case shows that the imaging characteristics of these rare tumors on contrast-enhanced CT combined with 3D-CT angiography can be helpful in reliably establishing a correct preoperative diagnosis.


2019 ◽  
Vol 17 (1) ◽  
Author(s):  
Chenglong Wang ◽  
Yijia Cao ◽  
Min Zeng ◽  
Lijuan Wang ◽  
Xiaojing Cao ◽  
...  

Abstract Background Lymph node inclusions are foci of ectopic tissue in lymph nodes, which were reported in different areas of the body. However, inclusions in the mediastinal lymph node are rare. Here, we report the first case of glandular inclusion within the parenchyma of the intrapulmonary lymph node in a patient with primary lung adenocarcinoma. Case presentation A computed tomography (CT) scan showed a solid pulmonary nodule in the right upper lobe in a 44-year-old man. After a fine needle aspiration biopsy diagnosis of adenocarcinoma, lobectomy and lymph dissection were performed. Histological sections of the lung demonstrated a papillary predominant adenocarcinoma and one intrapulmonary lymph node, which displayed glandular inclusion occupying the node parenchyma. The gland inclusion was very similar to metastasis, but was formed by two layers of epithelial cells, and the abluminal cells were positive for P63, P40, and CK5/6. The patient has remained alive without recurrence and metastasis at the last follow-up before publication. Conclusions It is very important to correctly diagnose a lymph node inclusion for proper clinical management.


2016 ◽  
Vol 2016 ◽  
pp. 1-4 ◽  
Author(s):  
Athanasia K. Papazafiropoulou ◽  
Angeliki M. Angelidi ◽  
Antonis A. Kousoulis ◽  
Georgios Christofilidis ◽  
Chariklia Sagia ◽  
...  

Introduction. Castleman’s disease (CD) is a rare lymphoproliferative disorder. CD is divided into two clinical subtypes: the most common unicentric and the less usual multicentric subtype. The majority of unicentric CD affects the mediastinum, while neck, abdomen, and axilla are less common locations.Case Presentation. Herein, we describe a rare case of unicentric CD in the right axilla in a 36-year-old white male with a medical history of hepatitis C virus infection admitted to our hospital due to palpation of a painless mass in the right axilla. Complete excision of the lesion was performed and, one year after the diagnosis, patient was free of the disease.Conclusions. Although infrequent, it is important to include CD in the differential diagnosis when evaluating axillary lymphadenopathy particularly in young patients with a low-grade inflammation process and chronic disease even in the absence of an abnormal blood picture or organomegaly.


2013 ◽  
Vol 3 (6) ◽  
pp. 509-511
Author(s):  
S Shrestha ◽  
U Nepal ◽  
N Lamichhane ◽  
P Chhetri

Castleman’s diseas is a rare lymphoproliferative disorder of unknown etiology. We report a 28 years old woman with solitary Castleman’s disease in the left pararenal space. This case was diagnosed preoperatively as renal cell carcinoma. The patient underwent a radical nephrectomy with dissection of pararenal mass. Histopathological examination of the surgically resected specimen showed the hyaline vascular type of Castleman’s disease. A preoperative diagnosis of Castleman’s disease is difficult; therefore, a surgical resection and histopathological evaluation can provide an accurate diagnosis of tumor. Taking this case into consideration, we suggest that Castleman’s disease should be included in the differential diagnosis of renal tumors. DOI: http://dx.doi.org/10.3126/jpn.v3i6.9004 Journal of Pathology of Nepal (2013) Vol. 3, 509-511


Author(s):  
Babu Manohar ◽  
Raees Abdurahiman

<p>Schwannomas are less common benign slow growing tumors originating from Schwann cells. In the head and neck region, schwannomas arise most commonly from the vagus nerve or the sympathetic chain. We present this case as the location of schwannoma is extremely rare and due to the diagnostic difficulties it posed. A 48 year old male presented with right neck swelling and breathing difficulty to our OPD. Patient underwent ultrasonogram of neck, MRI neck and Fine needle aspiration cytology (FNAC) of the lesion. Each of the investigations suggested different pathology which made the diagnosis challenging. During surgery, the lesion was found to arise from right recurrent laryngeal nerve. After excision of the lesion, the patient developed hoarse voice and the pathological examination revealed schwannoma. Schwannomas that originate from Schwann cells can affect any part of the body. MRI, CT, USG and FNAC have been suggested in the literature for diagnosing the lesion. Trucut biopsy should be considered in situations where FNAC becomes inconclusive. Surgical excision is the treatment of choice. Histologically, five variants of schwannomas have been described in the literature namely common, plexiform, cellular, epithelioid and ancient schwannoma. To conclude, schwannoma arising from RLN which masqueraded as a thyroid swelling is a rare entity. The diagnostic modalities suggested in the literature were unable to pin point the diagnosis. Once, FNAC shows an inadequate specimen, a trucut biopsy should be considered as the next investigation modality.</p>


Blood ◽  
2015 ◽  
Vol 126 (23) ◽  
pp. 5027-5027
Author(s):  
Nalini Hasija ◽  
Liliana Bustamante ◽  
Michael V. Jaglal ◽  
Lubomir Sokol

Background Human Immunodeficiency Virus (HIV)-negative Castleman's Disease (HIVnegCD) is a group of rare heterogeneous polyclonal lymphoproliferative disorders of poorly understood etiopathogenesis. Several histological subtypes were previously described including hyaline-vascular, plasma-cell, mixed and plasmablastic. Unicentric Castleman's disease (UCD) is usually treated with complete surgical excision of involved lymph node(s). Multicentric Castleman's Disease (MCD) frequently manifests with generalized lymphadenopathy and systemic B-symptoms due to inflammatory hypercytokinemia. An important role of inflammatory cytokine IL-6 was previously established in MCD in experimental and clinical studies. Patients with MCD usually require systemic therapy. Objectives Primary purpose of this study was to collect and analyze clinicopathological and laboratory characteristics and outcomes of patients with HIVnegCD treated in a single institution. Methods Institutional review board (IRB)-approved, Health Insurance Portability and Accountability Act (HIPAA) compliant, retrospective study was conducted at Moffitt Cancer Center. Clinical and laboratory characteristics were extracted from electronic medical records and analyzed using SPSS version 22.0 statistical software. Results We identified a total of 28 patients (pts) between 1993 and 2015. 14 pts had UCD and 14 MCD. Median age was 49 (35-87) years (UCD) and 47 (31 -79) years (MCD). Male: female ratio was 1:2.5 (UCD) and 1:1.33 (MCD). A majority of patients had a PS 0-1 in both groups (13/13 and 9/10 respectively). Only 2/13 (15.4%) pts with UCD presented with fatigue and 2 (15.4%) had CNS symptoms. 9 of 14 (64%) pts with MCD reported fatigue and 8/14 (57.1%) presented with night sweats and anorexia. 5 of 14 pts (36%) with MCD were concurrently diagnosed with POEMS syndrome, 2 pts with MGUS and 1 pt with smoldering IgA kappa light chain restricted multiple myeloma. HIV-1/2 ELISA test was negative in all tested pts (18/18). In the remaining 10 pts the test was not available, but a longitudinal follow-up revealed no evidence of immunodeficiency or history of opportunistic infections. HHV-8 test was negative in 9 of 9 tested pts. 2 of 18 pts showed polyclonal hypergammaglobulinemia. 10 of 18 pts had soluble IL-6 tested and only 1 pt with MCD had an elevated level. C-reactive protein (CRP) was elevated in only 1 of 10 pts. Confirmatory histology slide review in our institution revealed a hyaline-vascular subtype in 4 pts with UCD and 6 pts with MCD. 10 pts had plasma-cell subtype (6 with UCD and 4 with MCD) and 2 had mixed subtype (1 each in UCD and MCD). In 6 pts, pathological diagnosis was based only on outside report due to the absence of the original histology slides or insufficient tissue for confirmatory studies. 11 of 13 pts with UCD underwent surgical excision, 1 received radiation therapy and 1 chemotherapy in the frontline settings resulting in complete response. 1 patient with UCD received anti-IL-6 monoclonal antibody for the second line therapy followed by rituximab monotherapy. This patient was the only pt with UCD demonstrating progressive disease. Management of MCD included various systemic regimens listed in Table #1. Chemotherapy regimens included chloramubucil, thalidomide, cyclophosphamide, melphalan, DT-PACE, R-CHOP and lenalidomide. Only 3 pts died and all 3 had progressive refractory MCD. 2 of the pts who died also had POEMS syndrome. Median survival was not reached, as 25 of 28 pts were alive at the time of data collection. Conclusions This retrospective study indicated that HIVnegCD is a heterogeneous disorder with very good prognosis in patients with resectable UCD. Systemic therapy is usually required in patients with MCD. The increased frequency of malignancies associated with CD such as POEMS syndrome, multiple myeloma, and follicular dendritic cell sarcoma can have adverse impact on prognosis. Table 1. Treatment of MCD MCD First Line Surgery 1 12 8.3% Steroids 6 12 50% Rituximab 1 12 8.3% IL-6 2 12 16.7% Chemo1 2 12 16.7% Second Line Surgery 1 8 12.5% Rituximab 2 8 25% Chemo2 1 8 12.5% Combo chemo3 2 8 25% Surgery +XRT 1 8 12.5% Auto BMT 1 8 12.5% Third Line XRT 1 5 20% IL-6 1 5 20% Chemo4 2 5 40% Combo chemo5 1 5 20% 1 Chlorambucil/Pred, Thalidomide/Dex 2 Melphalan/Pred 3 Etoposide/Ritux, Lenalidomide/Ritux 4 DT-PACE, Cyclophosphamide/Pred 5 R-CHOP Disclosures Sokol: Janssen Research & Development, LLC: Consultancy.


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