EUS in the diagnosis of peripancreatic Castleman disease: A case report (with video).
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ABSTRACT:
SUBMITTER: Han Z
PROVIDER: S-EPMC10237606 | biostudies-literature | 2023 Mar-Apr
REPOSITORIES: biostudies-literature
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EUS in the diagnosis of peripancreatic Castleman disease: A case report (with video).
Han Ziying Z Han Xianlin X Li Yuan Y Feng Yunlu Y Zhang Shengyu S Yang Aiming A
Endoscopic ultrasound 20230301 2
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Project description:IntroductionCastleman disease (CD) is a lymphoproliferative disorder with lymph node hypertrophy. In the unicentric form (UCD), it affects one lymph node or chain of lymph nodes. In the multicentric form (DCM), there is hypertrophy of several lymph node chains with the formation of tumor masses, causing compressive symptoms. This case report showed a case of CD in a different location(inguinal region) associated to a multiple skin lesions.Presentation of the caseWe reported a UCD in a 43-year-old female patient with no previous comorbidities. Since January 2016, this patient developed erysipelas lesions of the left leg (LL) from the thigh root to the foot. Concomitantly, a tumor mass appeared in the inguinal region. In 2019 we performed a biopsy that revealed changes characteristic of CD. Due to extremely poor trophic conditions, the skin area with erysipelas was resected, and the raw surface was grafted.DiscussionAs an inference, the erysipelas may have been responsible for the subsequent lymphangitis, lymphedema and lymph node hypertrophy.ConclusionResection of the diseased skin and lymph node excision constitute the treatment of UCD and result in improvement of the clinical picture. Nevertheless, further study of the inflammatory reaction and of markers such as interleukin-6 and the presence of skin disorders in DC is needed.
| S-EPMC7393987 | biostudies-literature
Project description:Human herpes virus-8 (HHV8)-negative, idiopathic multicentric Castleman disease (iMCD) is a rare lymphoproliferative disorder sustained by pro-inflammatory cytokines, including interleukin-6 (IL-6). According to the international evidence-based criteria developed by the Castleman Disease Collaborative Network (CDCN), siltuximab, which works by inhibiting IL-6, is the recommended choice for iMCD treatment. We report a case of a 63-year-old white male with iMCD who has been on maintenance therapy with siltuximab for 15 years - representing one of the longest treatment periods of any patient with iMCD treated with siltuximab. The patient initially presented with fatigue and night sweats, with progressive worsening of the symptoms. Whole-body positron emission tomography/computed tomography revealed hypermetabolic lymphadenopathy. The patient had histopathologically confirmed Castleman disease, plasma cell type, and was negative for HHV8 and human immunodeficiency virus. The patient had abnormally high C-reactive protein (CRP) levels, a surrogate measure for IL-6. The patient was treated with high-dose steroids but had recurring lymphadenopathy early on. He was enrolled in the phase I dose-finding clinical trial of siltuximab, during which he achieved marked clinical improvement and sustained inhibition of CRP. The patient was enrolled in the long-term safety study and continues to receive siltuximab at 11 mg/kg every 3 weeks. He is presently receiving commercial siltuximab and has remained asymptomatic, with no evidence of lymphadenopathy. The case study presented is consistent with the evidence that siltuximab is a safe and effective therapy for the long-term management of iMCD. In addition, this case highlights the importance of prompt diagnosis for patients with iMCD, as effective therapy is available for patients as described in the CDCN and National Comprehensive Cancer Network iMCD treatment guidelines.Plain language summaryThe case of a 63-year-old white male with idiopathic multicentric Castleman disease who was successfully treated with siltuximab for 15 years: Idiopathic multicentric Castleman disease (iMCD) is a group of rare lymphoproliferative disorders with shared histopathological features that affect lymph nodes in multiple regions of the body. The signs and symptoms of iMCD can be varied, with the disease being mild in some patients while life-threatening in others. A timely diagnosis of iMCD can be challenging but is required for effective management. We report a case of a patient who was diagnosed with iMCD. The patient was given high-dose steroids but continued to show progressive disease. He was then started on siltuximab, a targeted antibody therapy against a specific cytokine (interleukin-6) involved in inflammation. The patient responded well to the treatment, has shown evidence of long-term disease control, and has not reported any serious adverse events related to long-term siltuximab use. He has received 11 mg/kg of siltuximab every 3 weeks for the past 15 years. This case emphasizes the value of using siltuximab therapy for long-term management of this rare disorder. In addition, it highlights the importance of prompt diagnosis for patients with iMCD, as effective therapy is available, as described in iMCD treatment guidelines.
| S-EPMC8894931 | biostudies-literature
Project description:BackgroundSystemic lupus erythematosus (SLE) is a systemic disease, which can bring damage to multiple organ systems. It is easily misdiagnosed as mechanical intestinal obstruction and treated by surgery, which not only brings physical pain to patients, but also increases their economic burden. On the other hand, Castleman disease (CD) is also a rare disease that can be easily missed clinically. Consequently, IPO in SLE complicated by CD is extremely rare in clinical practice and easily ignored for clinicians, which may result in delayed diagnosis, and treatment, and even overtreatment.Case descriptionAn 18-year-old Chinese woman presented with over a month's history of abdominal pain and fever, accompanied by abdominal distension and nausea. The patient was admitted to a local hospital before admission, and imaging test showed intestinal obstruction. After symptomatic treatment, abdominal pain was relieved, but symptoms reappeared about 20 days later. In addition, a red rash on face, light-sensitiveness and alopecia appeared 7 months prior to presentation. Physical examination showed a temperature of 38.9 ℃, facial butterfly erythema, enlarged axillary lymph nodes, lower abdominal tenderness, and diminished bowel sounds. Laboratory examinations showed proteinuria, decreased white blood cell, low C3 and low C4, positive antinuclear antibody (ANA), and positive anti-double-stranded DNA (anti-dsDNA). Abdominal noncontrast computed tomography (CT) showed partial small bowel obstruction. Chest contrast-enhanced CT showed multiple enlarged lymph nodes in the bilateral axillary and mediastinal areas. The results of the axillary lymph node biopsy were consistent with the typical histologic features of clear vascular CD. After glucocorticoid and immunosuppression therapy, the patient's immune indexes and proteinuria gradually returned to normal and abdominal pain did not recur during the follow up.ConclusionsIn order to avoid misdiagnosis of IPO in SLE and missed diagnosis of SLE complicated by CD, this case emphasizes the importance of medical history combined with appropriate laboratory examination, imaging examination and lymph node biopsy in SLE patients with lymphadenopathy for accurate diagnosis and reasonable treatment. At the same time, this case report aims to improve the diagnostic thinking of clinicians for similar patients.
| S-EPMC9652566 | biostudies-literature
Project description:Accurate diagnoses are crucial in determining the most effective treatment across different cancers. In challenging cases, morphology-based traditional pathology methods have important limitations, while molecular profiling can provide valuable information to guide clinical decisions. We present a 35-year female with lung cancer with choriocarcinoma features. Her disease involved the right lower lung, brain, and thoracic lymph nodes. The pathology from brain metastasis was reported as “metastatic choriocarcinoma” (a germ cell tumor) by local pathologists. She initiated carboplatin and etoposide, a regimen for choriocarcinoma. Subsequently, her case was assessed by pathologists from an academic cancer center, who gave the diagnosis of “adenocarcinoma with aberrant expression of β-hCG” and finally pathologists at our hospital, who gave the diagnosis of “poorly differentiated carcinoma with choriocarcinoma features”. Genomic profiling detected a KRAS G13R mutation and transcriptomics profiling was suggestive of lung origin. The patient was treated with carboplatin/paclitaxel/ipilimumab/nivolumab followed by consolidation radiation therapy. She had no evidence of progression to date, 13 months after the initial presentation. The molecular profiling could facilitate diagnosing of challenging cancer cases. In addition, chemoimmunotherapy and local consolidation radiation therapy may provide promising therapeutic options for patients with lung cancer exhibiting choriocarcinoma features.
2024-03-14 | GSE261551 | GEO
Project description: Not available
| S-EPMC7430900 | biostudies-literature
Project description:BACKGROUND:POEMS syndrome is a rare neoplastic syndrome reflected by plasma cell disorder. It is composed by polyneuropathy, organomegaly, endocrinopathy, monoclonal protein, and skin changes. It is also reported to associate with Castleman disease. The early identification and treatment are pivotal to reduce the morbidity and mortality. CASE PRESENTATION:Here we report a 66-year-old man with treated Castleman disease developing with sequential presence of endocrinopathy polyneuropathy, skin changes, organomegaly and extravascular volume overload within 18 years, which was finally confirmed as POEMS syndrome by positive monoclonal protein. He was thereafter successfully treated with prednisone and azathioprine as primary therapy and thalidomide as maintenance therapy. CONCLUSION:The diagnosis of POEMS is based on a cluster of disorder involved in varied organs. We report a rare case that triggers the need to consider POEMS syndrome diagnosis for patients carrying Castleman disease and polyneuropathy.
| S-EPMC6176504 | biostudies-other
Project description:RationaleAppendiceal intussusception is a rare disease. The definite preoperative diagnosis of appendiceal intussusception is rare and challenging. Here, we present a case of McSwain type V appendiceal intussusception in a 10-year-old boy. To our best knowledge, this is the first case report of a type V appendiceal intussusception that was preoperatively confirmed with sonography. Here, we have described in detail the ultrasound features and differential diagnosis of this rare disease.Patient concernsA 10-year-old boy presented with 3 days of recurrent intermittent mild abdominal pain. The result of ultrasonography suggested an ileocolic intussusception and a therapeutic air-contrast enema was requested to reduce the intussusception but failed at a local hospital.DiagnosesPhysical exam revealed mild tenderness in the lower right quadrant of the abdomen. However, ultrasonography showed a target-sign in cross section and a finger-like appearance in the longitudinal view. A diagnosis of McSwain type V appendiceal intussusception was made.InterventionsThe patient underwent an appendectomy after successful manual reduction on laparotomy. The appendix was successfully resected.OutcomesIntraoperatively, the appendix was completely inverted in the cecum, and the preoperative sonographic findings were confirmed. During follow-up, there were no signs of recurrence.LessonsPre-operatively, on ultrasound a type V appendiceal intussusception is usually misdiagnosed as an ileocolic intussusception. Radiologists must execute caution to avoid over reliance on the sonographic findings of intussusception, especially when there is a mismatch with clinical symptoms. It is especially important to accurately understand the surgical-anatomic configuration of type V appendiceal intussusception that creates a "target-sign" and a "finger-like" layout on ultrasonography.
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Project description:Biliary cystadenoma is a type of rare liver cystic tumor. Intrahepatic biliary cystadenomas are the most common, while extrahepatic biliary cystadenomas are rarely seen. Biliary cystadenoma tends to occur in middle-aged to older women and there is a lack of specific preoperative diagnostic markers. Recent advancements in technology and the development of the SpyGlass system have led to an increased use of cholangioscopy. Herein, we report a patient in whom a space-occupying lesion was found in the bile duct by SpyGlass, and who later underwent radical surgery. The pathology report indicated that the final diagnosis was biliary cystadenoma. SpyGlass cholangioscopy may be a novel and effective diagnostic method for biliary cystadenoma.
| S-EPMC10327893 | biostudies-literature
Project description: Not available
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