Capillary leak syndrome: initial presentation in a patient with ALK+ anaplastic large cell lymphoma associated with increased levels of serum cytokines

Umemoto, T; Watanabe, Tsutomu; Ogose, Takeshi; Kondo, Rieko; Nakatsu, Tadanori; Sakata, Akiyoshi; Yamashita, Michiko; Gotho, Tetsuya

doi:10.3109/10428194.2011.555893

Cited by 12 publications

(6 citation statements)

References 12 publications

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“…To the best of our knowledge, this case represents one of only two cases of ALCL reported that presented with SCLS [16]. Unlike most cases of SCLS, our patient did not have an IgG gammopathy.…”

Section: Discussionmentioning

confidence: 59%

Systemic Capillary Leak Syndrome as an Initial Presentation of ALK-Negative Anaplastic Large Cell Lymphoma

Lourdes

Al-Quran

Dang

et al. 2012

Case Reports in Hematology

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Systemic capillary leak syndrome (SCLS) is a rare disease characterized by third spacing of plasma into the extravascular compartment, leading to anasarca, hemoconcentration, and hypovolemic shock. It has been rarely associated with lymphomas, and reports usually indicate that it occurs after antineoplastic treatment. We present the case of a patient with ALK-negative anaplastic large cell lymphoma who presented with SCLS as the initial manifestation of her lymphoma. The SCLS resolved with treatment of the malignancy with steroids and chemotherapy.

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Section: Discussionmentioning

confidence: 59%

Systemic Capillary Leak Syndrome as an Initial Presentation of ALK-Negative Anaplastic Large Cell Lymphoma

Lourdes

Al-Quran

Dang

et al. 2012

Case Reports in Hematology

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“… 23 , 24 In an anaplastic lymphoma kinase (ALK) positive large cell lymphoma case complicated with CLS, IL-6 was elevated in the serum and was suggested to account for the fever in the patient. 13 However, these investigators did not test the cytokine level in the serous cavity effusion.…”

Section: Discussionmentioning

confidence: 99%

“… 8 For example, patients with lymphoma, such as those with diffuse large B-cell lymphoma or T-cell lymphoma, may develop CLS under cytokine therapy (e.g., interleukin (IL)-2, IL-11, interferon), bortezomib, rituximab, CAR-T therapy, and/or autologous stem cell transplantation. 9 – 14 In these patients, ascites and fever were commonly observed. 12 We report here a rare case of a patient with lymphoma who we believe is the first reported case of CLS with refractory ascites.…”

Section: Introductionmentioning

confidence: 92%

Capillary leak syndrome presenting as refractory ascites in a patient with lymphoma: A rare case report

Huang

Zhang

et al. 2023

J Int Med Res

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Capillary leak syndrome (CLS) is a serious disorder characterized by hypotension and refractory systemic oedema. CLS with marked ascites rather than systemic oedema is rare and prone to misdiagnosis and delayed treatment. We report here a case of marked ascites in an elderly male patient with hepatitis B virus reactivation. Following investigations to exclude common diseases that may have accounted for diffuse oedema and hypercoagulable state, anti-cirrhosis therapy failed and severe refractory shock developed 48 hours after admission. The patient developed mild pleural effusions followed by swelling of the face, neck, and extremities. A high cytokine concentration gradient was detected between serum and ascites. Peritoneal biopsy showed lymphoma cells. The final diagnosis was lymphoma recurrence complicated with CLS. Our case suggests that cytokine detection in serum and ascitic fluid may be helpful in the differential diagnosis of CLS. In similar cases, decisive intervention, such as, hemodiafiltration, should be implemented to lessen the likelihood of serious complications.

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“…In addition, hematological malignancies have shown to have a greater risk of SCLS and its associated mortality when compared with non-hematological malignancies, with the most common being non-Hodgkin lymphoma and multiple myeloma [24]. Incidentally, SCLS can be the presenting manifestation of a malignancy, e.g., in the literature, cases of anaplastic large cell lymphoma, large B-cell lymphoma, and T-cell lymphoma have been reported as initially presenting with SCLS [25][26][27]. Viral infections explicitly have been linked to SCLS including Ebola, dengue, Hantaviruses, Marburg, influenza A and more recently SARS-CoV-2 (severe acute respiratory syndrome coronavirus 2) [15].…”

Section: Discussionmentioning

confidence: 99%

Systemic capillary leak syndrome following granulocyte colony-stimulating factor therapy in a T-lymphoblastic leukemia/lymphoma patient: a case report

Sharie

Zu’bi

Sharie

et al. 2022

memo

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Introduction Systemic capillary leak syndrome (SCLS) is a rare and often fatal clinical entity used to describe a generalized increase in vascular permeability leading to fluid extravasation toward the interstitial compartment. SCLS could be an idiopathic disease or secondary to infections, malignancies or drugs. Case We present a case of presumably granulocyte colony-stimulating factor (G-CSF)-induced SCLS in a 21-year-old man diagnosed with T‑lymphoblastic leukemia/lymphoma. He received the 6th cycle (part B) of the hyper-CVAD chemotherapeutic regimen followed by the initiation of neutropenic fever prophylaxis protocol which included antibiotics and G‑CSF. In a course of hours, the patient became dyspneic, hypotensive, and edematous which required intensive care unit admission and was stabilized accordingly. In the following days the patient’s anasarca progressively increased which was associated with hypoalbuminemia, hypotension and anemia with pericardial and bilateral plural effusions. As a diagnosis of exclusion augmented by the acuity of such clinical event, observed concomitantly with the administration of the prophylaxis protocol, the suspicion of G‑CSF-induced SCLS was established. Consequently, G‑CSF was discontinued and treatment with dexamethasone and intravenous immunoglobulins (IVIG) was started. The patient’s condition improved significantly illustrated by hemodynamic stability in addition to improvement regarding the anasarca, hypoalbuminemia, and anemia. Follow-up scans suggest resolution of the pericardial and plural effusions. Conclusion SCLS remains a serios and potentially fatal complication of G‑CSF administration which should be taken into consideration, since such medication is widely utilized in oncology wards.

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Capillary leak syndrome: initial presentation in a patient with ALK+ anaplastic large cell lymphoma associated with increased levels of serum cytokines

Cited by 12 publications

References 12 publications

Systemic Capillary Leak Syndrome as an Initial Presentation of ALK-Negative Anaplastic Large Cell Lymphoma

Systemic Capillary Leak Syndrome as an Initial Presentation of ALK-Negative Anaplastic Large Cell Lymphoma

Capillary leak syndrome presenting as refractory ascites in a patient with lymphoma: A rare case report

Systemic capillary leak syndrome following granulocyte colony-stimulating factor therapy in a T-lymphoblastic leukemia/lymphoma patient: a case report

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