Gastric cancer complicated with hemophagocytic lymphohistiocytosis: case report and a brief review

Zhou, Yu-Shi; Cui, Yancheng; Yin, Ming; Xie, Qin; Shen, Zhanlong; Shi, Hongxia; Ye, Yingjiang; Liang, Bin

doi:10.21037/jgo-20-432

Cited by 6 publications

(5 citation statements)

References 29 publications

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“…In Table 1 , we report a comparison of a diagnosis with three other cases of secondary HLH in gastric cancer patients. In the case reported by Ramkumar ( 25 ) and in our case, the diagnosis was for a signet ring cell cancer, while we have no clear information about the histology of gastric cancer in the other two cases by Zhou ( 26 ). Thus, we cannot speculate about the risks of HLH related to histology.…”

Section: Discussioncontrasting

confidence: 67%

“…Again, in 2021, Zhou et al. from China ( 26 ), presented two cases of gastric cancer complicated with HLH. The patients received treatment including immunosuppressive agents immediately.…”

Section: Discussionmentioning

confidence: 99%

“…Thus, we cannot speculate about the risks of HLH related to histology. In the case from Ramkumar ( 25 ) and in one case from Zhou ( 26 ), the HLH “exploded” in a clinical manner after a gastrectomy; if one does not know about HLH, it seems strange to learn of deaths after gastric cancer resection. In our case, the patient had the metastatic disease as in the case presented by Ramkumar (supraclavicular lymphadenopathy), while in one case presented by Zhou, the patient had unresectable gastric adenocarcinoma.…”

Section: Discussionmentioning

confidence: 99%

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Hemophagocytic lymphohistiocytosis in gastric cancer: A rare syndrome for the oncologist. Case report and brief review

Monti

Marconi²,

Ambrosini-Spaltro³

et al. 2023

Front. Oncol.

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Hemophagocytic lymphohistiocytosis (HLH) is a rare and life-threatening condition characterized by uncontrolled activation of the immune system. HLH is a reactive mononuclear phagocytic response that occurs in association with a constellation of conditions such as malignancies and infections. The clinical diagnosis of HLH remains challenging because HLH can present with symptoms that significantly overlap with other causes of cytopenia, such as sepsis, autoimmune diseases, hematological cancers, and multiorgan failure. A 50-year-old man went to the emergency room (ER) for hyperchromic urine, melena, gingivorrhagia, and spontaneous abdominal wall hematomas. The first blood tests showed severe thrombocytopenia, alteration of the INR, and consumption of fibrinogen, and therefore, a diagnosis of disseminated intravascular coagulation (DIC) was made. A bone marrow aspirate showed numerous images of hemophagocytosis. With the suspicion of immune-mediated cytopenia, oral etoposide, intravenous immunoglobulin, and intravenous methylprednisolone were administered. Then, a diagnosis of gastric carcinoma was performed with a lymph node biopsy and gastroscopy. On the 30th day, the patient was transferred to the oncology ward of another hospital. On admission, he had serious piastrinopenia, anemia, hypertriglyceridemia, and hyperferritinemia. He was supported with a platelet transfusion and underwent a bone biopsy that showed a picture compatible with myelophthisis from diffuse medullary localization of a carcinoma of gastric origin. A diagnosis of HLH secondary to solid neoplasm was formulated. The patient started chemotherapy with oxaliplatin, calcium levofolinate, 5-fluorouracil bolus, 5-fluorouracil for 48 h (mFOLFOX6), and methylprednisolone. Six days after the third cycle of mFOLFOX6, the patient was discharged with the stabilization of his piastrinopenia condition. The patient continued chemotherapy with an improvement in his clinical conditions and normalization of hematological values. After 12 cycles of mFOLFOX, it was decided to start maintenance chemotherapy with capecitabine but, unfortunately, after only one cycle, HLH reappeared. The oncologist has to keep in mind the existence of HLH when there is an unusual clinical presentation of cancer, such as cytopenia affecting ≥2 lineages and alterations of ferritin and triglycerides other than fibrinogen and coagulation. Increased attention and additional research as well as a close collaboration with hematologists are needed to benefit patients with solid tumors complicated by HLH.

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Section: Discussioncontrasting

confidence: 67%

“…Again, in 2021, Zhou et al. from China ( 26 ), presented two cases of gastric cancer complicated with HLH. The patients received treatment including immunosuppressive agents immediately.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Hemophagocytic lymphohistiocytosis in gastric cancer: A rare syndrome for the oncologist. Case report and brief review

Monti

Marconi²,

Ambrosini-Spaltro³

et al. 2023

Front. Oncol.

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“…M-HLH is common in hematologic tumors, such as T-cell or NK-cell lymphomas or leukemias, followed by B-cell lymphomas, and HLH caused by solid tumors is rare ( 20 ). There have been numerous previous reports on M-HLH, such as gastric cancer ( 21 ), glioblastoma ( 22 ), prostate cancer ( 23 ), and pembrolizumab in the treatment of head and neck squamous cell carcinoma to induce HLH ( 24 ). Here we report a case of intracranial germinoma causing HLH.…”

Section: Discussionmentioning

confidence: 99%

Hemophagocytic lymphohistiocytosis during treatment of intracranial multifocal germinoma: a case report and literature review

Guo,

Liu,

Chen

et al. 2024

Front. Oncol.

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Hemophagocytic lymphohistiocytosis (HLH), also known as hemophagocytic syndrome (HPS), is a benign histiocytosis with hyperreactive proliferation of the mononuclear phagocyte system caused by immune function abnormalities, which often occurs under the background of genetic mutations, inflammation, infection or tumors. Because the research on malignancy-associated HLH (M-HLH) is focused on hematological malignancies, reports on HLH secondary to solid tumors are rare. In this case, we report a 14-year-old girl who developed HLH during treatment for intracranial multifocal germinoma, and the disease was controlled after hormone combined with etoposide(VP-16) and other related treatments. To our knowledge, there have been no documented cases of HLH caused by intracranial multifocal germinoma.

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“…with sHLH. In a recent report from China, two gastric cancer patients with sHLH were treated with glucocorticoids plus etoposide and etoposide/doxorubicin, respectively, but after clinical recovery sHLH relapsed [7]. As IL-1 is crucial in the pathogenesis of HLH, we opted for subcutaneous application of anakinra-no adverse events occured and our patient quickly recovered from sHLH.…”

mentioning

confidence: 96%

Progressive Thrombocytopenia and Clinical Deterioration with Initial Diagnosis of Esophageal Cancer-Secondary Hemophagocytic Lymphohistiocytosis as a Complicating Cause

Nassir¹

2022

GMR

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A 52-year-old male patient presented in the emergency department with fatigue and persistent back and flank pain. Computed tomography revealed lymphadenopathy and disseminated osseous lesions. Staging was completed and a low differentiated signet ring cell carcinoma of the distal esophagus (AEG I), UICC stage IV [1], was diagnosed. Immunohistochemistry showed expression of human epidermal growth factor receptor 2 (HER2)/neu and E-cadherin without expression of programmed death-ligand [1]. No microsatellite instability was detected. Palliative polychemotherapy with 5-fluorouracil, folinic acid, oxaliplatin and docetaxel (FLOT) was initiated. Due to progressive pancytopenia with transfusion-dependent thrombocytopenia, abnormal renal function tests, fever and rapid clinical deterioration, we suspected malignancy-associated Thrombotic Microangiopathy (TMA), and therefore promptly initiated daily plasmapheresis. However, no objective response upon plasmapheresis was observed, while extended laboratory tests revealed elevated levels of lactate dehydrogenase (1477U/l, range: 135-250U/l), ferritin (4410µg/l, range 30-400µg/l), triglycerides (366mg/dl, range >200mg/dl) and soluble interleukin 1 receptor (1807IU/ml, range: <710IU/ml). Abdominal ultrasonography revealed splenomegaly. Notably, bone marrow biopsy revealed extensive carcinomatosis and activation of stromal macrophages (Figure 1). According to those findings and a calculated H-score (reference: http://saintantoine.aphp.fr/score/), malignancy associated Secondary Hemophagocytic Lymphohistiocytosis (sHLH) was diagnosed. Thus, we initiated treatment with interleukin (IL)-1 inhibition (Anakinra), prednisolone and continuation of polychemotherapy, resulting in a significant clinical improvement and decline of ferritin (1990.4µg/l) and lactate dehydrogenase (860U/l) levels. Subsequently, the patient successfully completed six cycles of polychemotherapy but eventually died due to progressive esophageal carcinoma. What do we learn from this unusual case? First, distinguishing sHLH from TMA in the context of rapid onset thrombocytopenia along with clinical and laboratory deterioration is extremely challenging. However, extensively elevated ferritin levels are indicative for HLH and may guide diagnostic considerations early on [2]. Second, Bone Marrow Carcinomatosis (BMC) due to gastric or esophageal carcinoma is a highly rare event [3,4], and concomitant sHLH is a yet underreported, but potentially life-threatening phenomenon in this context. HLH is a hyperinflammatory syndrome leading to an uncontrolled cytokine storm with an all-cause mortality of approximately 40% in adults [5]. Although exact path mechanisms remain unclear, dysregulation of immune homeostasis due to chemotherapy and underlying malignancy might lower the threshold for triggering sHLH [6]. However, in this patient we considered BMC as driver of sHLH and therefore decided to continue chemotherapy. Third, therapeutic decision-making remains challenging due to a lack of validated treatment protocols in a...

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Gastric cancer complicated with hemophagocytic lymphohistiocytosis: case report and a brief review

Cited by 6 publications

References 29 publications

Hemophagocytic lymphohistiocytosis in gastric cancer: A rare syndrome for the oncologist. Case report and brief review

Hemophagocytic lymphohistiocytosis in gastric cancer: A rare syndrome for the oncologist. Case report and brief review

Hemophagocytic lymphohistiocytosis during treatment of intracranial multifocal germinoma: a case report and literature review

Progressive Thrombocytopenia and Clinical Deterioration with Initial Diagnosis of Esophageal Cancer-Secondary Hemophagocytic Lymphohistiocytosis as a Complicating Cause

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