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Liu W, Tao Z, Liang R, Hu X. Castleman disease complicated by rheumatoid arthritis and postoperative chylous leakage: A case report. Medicine (Baltimore) 2025; 104:e41559. [PMID: 39960936 PMCID: PMC11835113 DOI: 10.1097/md.0000000000041559] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/18/2024] [Accepted: 01/30/2025] [Indexed: 02/20/2025] Open
Abstract
RATIONALE Castleman disease (CD) is a rare disorder characterized by nonspecific clinical presentations and imaging findings, making it prone to misdiagnosis and missed diagnosis. This report details the diagnosis and treatment process of a patient with CD complicated by rheumatoid arthritis (RA) who developed chylous leakage postoperatively, highlighting the potential risks of infection and nutritional depletion, aiming to enhance the understanding of CD. PATIENT CONCERNS A patient with CD complicated by RA developed chylous leakage postoperatively, which posed risks of infection and nutritional depletion. DIAGNOSES The patient underwent a left axillary lymph node excisional biopsy on November 13, 2019, and was diagnosed with HV-CD based on pathology. INTERVENTIONS No further axillary lymph node dissection or adjuvant therapy was performed. Postoperatively, the patient developed significant chylous leakage from the biopsy incision, which resolved after 2 weeks of drainage, dietary modifications, and anti-infective treatment. OUTCOMES Follow-up showed no disease progression, and the chylous leakage resolved with appropriate management. LESSONS A history of RA may be associated with the onset of CD. HV-CD generally has a favorable prognosis, and chylous leakage post-axillary lymph node biopsy, though rare, can be effectively managed with appropriate drainage, dietary control, and infection prevention.
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Affiliation(s)
- Wei Liu
- Department of Breast, Jinan University Affiliated Guangzhou Red Cross Hospital: Guangzhou Red Cross Hospital, Guangzhou, Guangdong, China
| | - Zhuoyan Tao
- Department of Breast Surgery, Shenzhen Futian District Maternal and Child Health Care Hospital, Shenzhen, Guangdong, China
| | - Rong Liang
- Department of Breast, Jinan University Affiliated Guangzhou Red Cross Hospital: Guangzhou Red Cross Hospital, Guangzhou, Guangdong, China
| | - Xinpeng Hu
- Department of Breast Surgery, The Affiliated Traditional Chinese Medicine Hospital of Guangzhou Medical University, Guangzhou, China
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Patil MB, Sindgikar VU, Kulkarni S, Biradar D, Siddesh A. Castleman's Disease Presenting as an Unusual Pelvic Retroperitoneal Mass. Cureus 2024; 16:e72196. [PMID: 39583477 PMCID: PMC11584245 DOI: 10.7759/cureus.72196] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/10/2024] [Accepted: 10/23/2024] [Indexed: 11/26/2024] Open
Abstract
Castleman's disease (CD) is characterised by benign lymphoepithelial proliferation and is a peculiar form of angiofollicular lymph node hyperplasia rather than a neoplasm or a hamartoma. CD is broadly classified as unicentric CD (UCD) and multicentric CD. In the unicentric variant, patients have localised disease affecting only a single lymph node or a group of adjacent nodes in a single region, which clinically presents as an enlarging mass without any other significant symptoms. The mediastinum and thoracic lymph nodes are commonly involved. However, it is uncommon for CD to occur in the pelvic presacral region of the retroperitoneum. A 31-year-old male presented with progressive, dull aching, intermittent lower abdomen pain for three months with no aggravating or relieving factors or any associated symptoms. He was haemodynamically stable, and his general physical examination was normal. An abdominal examination elicited mild tenderness in the left iliac region of the abdomen. Contrast-enhanced computed tomography of the abdomen and pelvis revealed an encapsulated nodal mass with intralesional calcification displacing the bladder to the right side, and fine needle aspiration cytology showed atypical cells. An exploratory laparotomy was performed with an in toto excision biopsy of the pelvic retroperitoneal mass, and it was diagnosed as a hyaline-vascular variant of UCD. Immunohistochemistry revealed angiofollicular hyperplasia and atretic germinal centres that are crossed by sclerotic vessels and hyalinisation, confirming the diagnosis. The patient is currently asymptomatic and is leading a routine life. The hyaline-vascular variant is the most common variant seen in UCD. Compared to the multicentric type, the unicentric type seldom exhibits systemic involvement. Surgical resection is typically curative. Although it is asymptomatic, it is essential to achieve complete surgical resection to prevent the neoplastic potentialities of CD. In the presence of an uncertain solitary solid pelvic retroperitoneal mass, the diagnosis of UCD should be considered, as surgical resection can achieve a favourable outcome.
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Affiliation(s)
- Mallikarjun B Patil
- General Surgery, Shri B. M. Patil Medical College Hospital and Research Centre, BLDE (Deemed to be University), Vijayapura, IND
| | - Vikram U Sindgikar
- General Surgery, Shri B. M. Patil Medical College Hospital and Research Centre, BLDE (Deemed to be University), Vijayapura, IND
| | - Sai Kulkarni
- Pathology, Shri B. M. Patil Medical College Hospital and Research Centre, BLDE (Deemed to be University), Vijayapura, IND
| | - Dayanand Biradar
- General Surgery, Shri B. M. Patil Medical College Hospital and Research Centre, BLDE (Deemed to be University), Vijayapura, IND
| | - Ashwin Siddesh
- General Surgery, Shri B. M. Patil Medical College Hospital and Research Centre, BLDE (Deemed to be University), Vijayapura, IND
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K A M, Kota SR, Mudigonda N, Kumar G, Abuji K. Mesenteric Castleman Disease Mimicking Neuroendocrine Tumor. Cureus 2024; 16:e61549. [PMID: 38962652 PMCID: PMC11219578 DOI: 10.7759/cureus.61549] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/31/2024] [Indexed: 07/05/2024] Open
Abstract
Castleman disease (CD) is a benign lymphoproliferative disorder of unknown etiology, which can involve any part of the body. CD can involve a single lymph node (unicentric) or multiple lymph nodes (multicentric) of which unicentric CD is the most common type. The unicentric CD is usually localized, asymptomatic, and often appears as an incidental mass on radiographs, whereas multicentric CD is characterized by systemic involvement. Mesenteric involvement of CD is very rare. In this article, we present a case of the unicentric CD of small bowel mesentery, which mimicked a neuroendocrine tumor preoperatively.
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Affiliation(s)
- Manjesh K A
- General Surgery, ESIC (Employee's State Insurance Corporation) Medical College, Hyderabad, IND
| | - Sreedhar Rao Kota
- General Surgery, ESIC (Employee's State Insurance Corporation) Medical College, Hyderabad, IND
| | - Narender Mudigonda
- General Surgery, ESIC (Employee's State Insurance Corporation) Medical College, Hyderabad, IND
| | - Gokul Kumar
- General Surgery, ESIC (Employee's State Insurance Corporation) Hospital, Hyderabad, IND
| | - Kishore Abuji
- General Surgery, ESIC (Employee's State Insurance Corporation) Medical College, Hyderabad, IND
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Shah RH, Cioffi-Lavina M, Jaguan A, Varella M. Unusual Presentation of Castleman's Disease Encroaching on the Brachial Plexus. Cureus 2021; 13:e16981. [PMID: 34527461 PMCID: PMC8421014 DOI: 10.7759/cureus.16981] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/28/2020] [Accepted: 08/07/2021] [Indexed: 11/26/2022] Open
Abstract
Castleman's disease is an uncommon benign lymphoproliferative disorder that commonly involves the mediastinum. We report an unusual case that involves the presentation of unicentric Castleman's disease in a 52-year-old female. The patient had a supraclavicular mass extending onto the brachial plexus. The approach to the treatment and plan for supraclavicular masses is complex due in part to the extensive list of differential diagnoses possible. In this case specifically, while the mass was ultimately determined to be benign, post-surgery, the location of the mass intraoperatively made for a very technically challenging and complex dissection. Post surgical resection, the patient reported no complications. This case highlights the importance of clinical judgement, imaging and surgical technique in removing a mass encroaching on the brachial plexus.
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Affiliation(s)
- Rahil H Shah
- Internal Medicine, Jackson Memorial Hospital/University of Miami, Miami, USA
| | | | - Abraham Jaguan
- Department of Otolaryngology, Mercy Hospital, Miami, USA
| | - Marcia Varella
- Department of Medical and Population Health Sciences Research, Florida International University Herbert Wertheim College of Medicine, Miami, USA
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Bradai S, Khsiba A, Nakhli A, Mahmoudi M, Mohamed AB, Chaabane A, Chelbi E, Mona M, Hamzaoui L, Azouz M. Abdominal unicentric Castleman's disease: a case report. Pan Afr Med J 2021; 38:339. [PMID: 34285761 PMCID: PMC8265241 DOI: 10.11604/pamj.2021.38.339.28859] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/13/2021] [Accepted: 03/28/2021] [Indexed: 11/11/2022] Open
Abstract
Castleman's disease is a rare disease characterized by benign lymphoepithelial proliferation. There are two forms: unicentric and multicentric Castleman's disease. Mediastinal location is the most frequent. Intra-abdominal Castleman's disease is a rare presentation. We report a case of 65-year-old female who presented with epigastric pain. Investigations revealed a retroperitoneal mass which was surgically resected. Histopathological examination showed hyaline-vascular type Castleman's disease. In conclusion, Castelman´s disease is a diagnostic challenge and it must be included in the differential diagnosis of retroperitoneal tumors.
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Affiliation(s)
- Samir Bradai
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Amal Khsiba
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Abdelwaheb Nakhli
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Moufida Mahmoudi
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Asma Ben Mohamed
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Abir Chaabane
- Histopathology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Emna Chelbi
- Histopathology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Medhioub Mona
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Lamine Hamzaoui
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
| | - Mousadek Azouz
- Gastroenterology Department, Mohamed Tahar Maamouri Hospital, Nabeul, Tunisia
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Bhogal RH, Wotherspoon A, Khan AK. Mesenteric Castleman's disease mimicking neuroendocrine tumour. Int J Surg Case Rep 2019; 63:56-58. [PMID: 31563665 PMCID: PMC6796708 DOI: 10.1016/j.ijscr.2019.09.002] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/25/2018] [Revised: 08/02/2019] [Accepted: 09/04/2019] [Indexed: 11/20/2022] Open
Abstract
Mesenteric Castleman’s disease is a rare pathology. It should be included in the differential for abdominal lymphadenopathy. Patients have an excellent prognosis after resection. Introduction Castleman’s disease is a rare entity and is a benign hyperplastic enlargement of lymph nodes. It can occur anywhere within the body but only approximately 50 cases of mesenteric Castleman’s disease have been reported within the literature. Case presentation We report a female patient who was thought to have developed a neuroendocrine tumour within the small bowel and a large nodal mass within the mesentery based on nuclear scintigraphy imaging. Following surgical resection the nodal mass found confirmed to be Castleman’s disease. Discussion Although rare complete resection of mesenteric Castleman’s disease offer excellent long term outcome although the diagnosis may only be made after surgery. Conclusion The diagnosis of mesenteric Castleman’s disease is often made post-operatively but complete surgical resection offers excellent long-term survival.
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Affiliation(s)
- Ricky H Bhogal
- Department of Academic Surgery, The Royal Marsden Hospital, Fulham Road, London, SW3 6JJ, United Kingdom.
| | - Andrew Wotherspoon
- Department of Histopathology, The Royal Marsden Hospital, Fulham Road, London, SW3 6JJ, United Kingdom
| | - Aamir K Khan
- Department of Academic Surgery, The Royal Marsden Hospital, Fulham Road, London, SW3 6JJ, United Kingdom
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Zhai B, Ren HY, Li WD, Reddy S, Zhang SJ, Sun XY. Castleman disease presenting with jaundice: A case report and review of literature. World J Clin Cases 2019; 7:373-381. [PMID: 30746379 PMCID: PMC6369394 DOI: 10.12998/wjcc.v7.i3.373] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/30/2018] [Revised: 11/26/2018] [Accepted: 12/12/2018] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Castleman disease (CD) is a rare lymphoproliferative disorder that presents with various symptoms. CD accompanied with jaundice is uncommon since there are only 11 cases reported in the literature. CASE SUMMARY Here we report a 62-year-old woman who was admitted to the hospital with signs and symptoms of intermittent jaundice. Biochemical tests showed higher serum levels of total and direct bilirubin, and normal serum levels of tumor markers and interleukin-6. Contrast-enhanced computed tomography detected a 6 cm × 4 cm × 2.5 cm mass between the hepatoduodenal ligament and the inferior vena cava. The mass was successfully excised and the patient had a complete resolution of symptoms. A diagnosis of idiopathic unicentric CD was made based upon histological examination, which demonstrated the pathological features of CD of mixed type, including hyperplasia of follicular lymphoids with abundant plasma cells, degenerative germinal centers, interfollicular vascularity and hyaline degeneration. The diagnosis was corroborated by immunohistochemical analysis which detected multiple biomarkers. CONCLUSION This is the first study that describes the clinicopathological features of CD presenting with jaundice, which may deepen and extend our understanding of this disease.
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Affiliation(s)
- Bo Zhai
- Department of General Surgery, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
- Hepatosplenic Surgery Center, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Hai-Yang Ren
- Department of General Surgery, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Wei-Dong Li
- Department of General Surgery, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
- Hepatosplenic Surgery Center, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Shiva Reddy
- Department of Molecular Medicine and Pathology, Faculty of Medical and Health Sciences, The University of Auckland, Auckland 1142, New Zealand
| | - Shu-Jun Zhang
- Department of Pathology, The Fourth Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
| | - Xue-Ying Sun
- Hepatosplenic Surgery Center, The First Affiliated Hospital of Harbin Medical University, Harbin 150001, Heilongjiang Province, China
- Department of Molecular Medicine and Pathology, Faculty of Medical and Health Sciences, The University of Auckland, Auckland 1142, New Zealand
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