Bicytopenia and leukoerythroblastosis: a rare initial presentation of signet ring cell gastric adenocarcinoma

Takayasu, Vilma; Goto, Edna Harumi; Casagrande, Mayra Zanon; Miranda, P.; Diniz, Gabriela Bezerra de Freitas; Monteiro, Micaela Frasson; Felipe-Silva, Aloísio

doi:10.4322/acr.2017.014

Cited by 10 publications

(13 citation statements)

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“…Signet-ring cells have a characteristic morphological appearance with large clear vacuolated cytoplasmic inclusions displacing the nucleus to the periphery. [ 1 – 24 ] Such morphological appearance is well recognized in lymph nodes from patients with metastatic adenocarcinoma, but rarely seen in bone marrow biopsies. According to previous reports, the most common site of origin of metastatic adenocarcinoma with signet-ring cell morphology involving the bone marrow is the gastrointestinal tract.…”

Section: Discussionmentioning

confidence: 99%

“…Among these cases, gastrointestinal tract sites such as gastric, small bowel, ampulla of vater, and intrahepatic bile duct are the more common sites of origin. [ 1 – 24 ] However, it should be noted that hematolymphoid neoplasms can also present with signet-ring cell morphology which may lead to diagnostic pitfalls. [ 24 – 28 ] Here, we present a very unusual case of a 67-year-old female with invasive lobular breast carcinoma, confirmed by breast biopsy and histopathologic examination.…”

Section: Introductionmentioning

confidence: 99%

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Signet-ring cells in the bone marrow as an indication of cryptic metastasis of breast carcinoma

Leckey

Zhang

et al. 2019

Medicine

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Rationale:Signet-ring cell is a rare morphological finding in bone marrow, which usually indicates metastatic carcinoma from either the gastrointestinal tract or a primary hematolymphoid neoplasm. Here, we present a very unusual case of lobular breast carcinoma with metastasis to the bone marrow.Patient concerns:A 67-year-old female with estrogen receptor (ER)-positive lobular breast carcinoma was staged as T3N3M0, and treated with modified radical mastectomy followed by chemotherapy and radiotherapy. One year after treatment, she was noted to have moderate thrombocytopenia on complete blood count with the remainder of the parameters within normal limits. Radiographic examination revealed no evidence of recurrent disease.Diagnosis:Bone marrow biopsy was performed to exclude therapy-related myelodysplastic syndrome (MDS), which demonstrated hypercellularity with “hyperplastic” hematopoiesis. Upon closer inspection, a few signet-ring cells were identified which morphologically resembled histiocytes. These formed an interstitial infiltrate among the predominantly hematopoietic elements, and could have been easily overlooked. Immunohistochemistry demonstrated that these signet-ring cells were positive for pancytokeratin as well as ER which confirmed metastatic lobular breast carcinoma. On retrospective review of the aspirate smear, rare signet-ring cells were identified.Interventions:The patient was treated with additional chemotherapy.Outcomes:The patient eventually succumbed to overt dissemination after 14 months.Lessons:Due to the relative discohesiveness of lobular breast carcinoma, the cells frequently assume single-cell infiltration in bone marrow. This attribute, along with small cell size, bland cytologic features and paucity of tissue response, contributes to its escaping from identification on hematoxylin-eosin (H&E) sections. In this case, the signet-ring cells were hidden in apparently hyperplastic hematopoiesis. Careful inspection raised the possibility of occult metastasis which was readily detected and confirmed with immunohistochemistry.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Signet-ring cells in the bone marrow as an indication of cryptic metastasis of breast carcinoma

Leckey

Zhang

et al. 2019

Medicine

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“…The refractory anemia of our patient was assumed as a result of the impaired hematopoiesis (bone marrow infiltration) and the continuous bleeding by the neoplastic gastric lesion. 12 Prognosis of patients with bone marrow disseminated carcinomatosis associated with gastric cancer is poor. Chemotherapy is the only effective treatment.…”

Section: Discussionmentioning

confidence: 99%

An unusual hematological presentation of gastric adenocarcinoma

Koncoro¹,

Setijoso²,

Prasetiawaty³

et al. 2018

GHOA

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Introduction: Gastric malignancy remains a lethal disease, especially in developing countries. Advanced disease mostly involve peritoneum, liver, and lung. Bone involvement is rare, and bone marrow dissemination is even rarer. Hematological abnormalities may be seen as complications of gastric malignancy. Therefore knowledge of gastric malignancy possibilities spreading to bone marrow and causing hematological complications need to be taken. Case illustration:This case report describes a 33-year-old woman who complained of weakness, waxing and waning fever, leg pain, back pain, and weight loss. The patient examination revealed pallor, and the laboratory work-up depicted severe anemia and thrombocytopenia; the peripheral blood smear was consistent with leukoerythroblastosis. Bone marrow biopsy revealed adenocarcinomas. During hospitalization, the patient presented melena, and an upper digestive endoscopy showed an ulcerated and infiltrative lesion in antrum which the histopathology reported evidence of gastric adenocarcinoma. PET-CT scan showed multiple metastatic of lymph node in neck, chest, and abdomen. Liver and bone also showed diffuse metastases. Discussion:Our case highlights an unusual case of gastric adenocarcinoma with manifestations of bicytopenias and leukoerythroblastosis. As the diagnosis of gastric adenocarcinoma is based on endoscopic biopsy, the diagnosis can be difficult if the complaints is nonspecific, particularly in advanced stage. Diagnosis also become difficult in patients with bicytopenia and it is important to have a broad differential diagnosis when approaching a patient with bone pain. The aggressive behavior of this malignancy made poor outcome on patient.

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“…Isolated leukoerythroblastosis resembling leukemia may also be seen in severe infections [ 10 ]. However, leukoerythroblastosis with cytopenias (usually bi- and pancytopenias) is often indicative of BM infiltration and is more frequently an initial presentation of solid malignancies, like gastric adenocarcinoma [ 11 ], or signet ring cell poorly differentiated carcinoma [ 12 ]. In such circumstances, a diagnosis of non-hematological malignancy may be achieved through analysis of a BM biopsy, such as has been described in a case of metastatic lobular breast cancer by our group and others [ 13 , 14 ].…”

Section: Discussionmentioning

confidence: 99%

Leukoerythroblastosis with Cytopenia as an Initial Presentation of Lung Adenocarcinoma

2018

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A 74-year-old male with a history of chronic lymphocytic leukemia (CLL) previously treated with fludarabine/cyclophosphamide/rituximab (FCR) 5 years ago, presented with progressive fatigue, mucocutaneous bleeding, and cytopenias (hemoglobin 51 g/L, platelets 8.0 × 10⁹/L, lymphocytes 0.4 × 10⁹/L). He had normal respiratory findings, and no lymphadenopathy or hepatosplenomegaly. Further workup revealed a small spiculated lung nodule and multiple sclerotic bony lesions. Due to bleeding/profound thrombocytopenia, lung biopsy was not feasible. Peripheral smear revealed leukoerythroblastosis with few nucleated red blood cells and left shift of granulocytes. Bone marrow (BM) aspirate yielded a dry tap with clusters of extrinsic atypical cells on touch preparations. BM core biopsy showed infiltration and near complete replacement by a population of highly atypical cells with surrounding fibrosis. Cells were positive for cytokeratins CK7 and CK8/18, Napsin A, and thyroid transcription factor-1, specific for a primary poorly differentiated lung adenocarcinoma. Leukoerythroblastosis in association with cytopenia often indicates a BM infiltration and warrants an early BM biopsy to rule out hematological and solid malignancies, particularly in CLL patients treated with FCR. In our case, a diagnosis of a lung adenocarcinoma was established by BM examination, the only clinically feasible diagnostic modality.

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Bicytopenia and leukoerythroblastosis: a rare initial presentation of signet ring cell gastric adenocarcinoma

Cited by 10 publications

References 20 publications

Signet-ring cells in the bone marrow as an indication of cryptic metastasis of breast carcinoma

Signet-ring cells in the bone marrow as an indication of cryptic metastasis of breast carcinoma

An unusual hematological presentation of gastric adenocarcinoma

Leukoerythroblastosis with Cytopenia as an Initial Presentation of Lung Adenocarcinoma

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