G‑CSF producing oral carcinoma with diffuse uptake of FDG in the bone marrow: A case report

Abstract

Abstract.A 78-year-old male patient was referred to the Department of Oral Surgery, Hokuto Hospital (Obihiro, Japan) for painless swelling on the left neck and tongue. Histopathological examination of a biopsy specimen resulted in a diagnosis of squamous cell carcinoma of the tongue. Imaging examinations revealed cervical lymph node metastases on both sides, along with diffuse uptake of 18 F-fluorodeoxyglucose (FDG) in the bone marrow of the spine and pelvis. Hematologic tests revealed an increased white blood cell (WBC) count and serum concentrations of granulocyte colony stimulating factor (G-CSF). These findings suggested a G-CSF producing tumor, with fluctuations of WBC count, serum G-CSF concentration, and FDG uptake in the bone marrow, associated with tumor shrinkage and enlargement, an indicator of tumor status. IntroductionGranulocyte colony stimulating factor (G-CSF) is a cytokine mainly produced by macrophages, fibroblasts and endothelial cells in an inflammatory milieu. G-CSF stimulates neutrophil precursors, resulting in an increase in neutrophils, and recruits neutrophils from the bone marrow to peripheral blood. G-CSF is an important factor in infection prophylaxis, and recombinant G-CSF is universally used to treat neutropenia (1). G-CSF is also produced by non-hematologic malignancies with high leukocyte counts, consisting predominantly of neutrophils, in patients without infectious diseases. Most of these G-CSF producing tumors are present in the lungs (2), with tumors in the oral regions being rare. This report describes a patient with a tongue carcinoma producing G-CSF, as well as showing diffuse uptake of FDG in the bone marrow. Case reportIn July 2013, a 78-year-old man visited the Department of Oral Surgery, Hokuto Hospital (Obihiro, Japan) with a 2-week history of painless swelling on the left neck and tongue. The patient had no systemic complications and no significant family history. Some cervical lymph nodes on both sides were palpable (Fig. 1A). Intra-oral examination showed a tumor with induration about 40 mm in diameter on the left side of the tongue (Fig. 1B). Cytological examination of a swollen left lymph node showed atypical squamous cells, and pathological examination of a biopsy of the tongue tumor revealed a squamous cell carcinoma. A computed tomography (CT) scan with contrast demonstrated a large lateral oral tongue tumor of diameter 42 mm, without extension to the extrinsic muscles of the tongue; and some metastatic cervical lymph nodes that were enlarged, nonhomogeneously enhanced, and partially necrotic. Metastatic disease of left middle jugular lymph node was >30 mm in maximum diameter (Fig. 2). 18 F-fluorodeoxyglucose-positron emission tomography (FDG-PET)/CT showed abnormally high uptake by the tongue tumor (maximum standardized uptake value [SUVmax] 22.19) and by the four large metastatic nodes, with the large left middle jugular node having an SUVmax of 14.43 (Fig. 3A). Diffuse FDG uptake was also observed in the bone marrow of the spine and pelvis (Fig. 3B). These f...

Granulocyte Colony-stimulating Factor Producing Oropharyngeal Squamous Cell Carcinoma

“…In most studies, G CSF was immunohistochemically confined to tumour cells. However, one study reported a negative result for G-CSF immunohistochemical staining (9) and in our study, as well as some of the tumour cells, some of the infiltrating leukocytes stained positiveIy for G-CSF. There has been speculation that the rapid secretion of G-CSF from tumour cells may lead to negative staining results (9).…”

Case Report: Asymmetric Bone Marrow Involvement in Patients With Acute Leukemia After Allogeneic Hematopoietic Stem Cell Transplantation

“…In a retrospective study of 28 children suspected of leukemia progression or recurrence during / after chemotherapy or allo-HSCT, 14 cases with positive 18 F-FDG-PET/CT scans were associated with increased blasts in bone marrow biopsies, and the mean SUV max was significantly higher than what is seen with infectious diseases ( 15 ). In addition, G-CSF induced high uptake of FDG in bone marrow system was always diffusely distributed ( 16 , 17 ). In the second case, the adjacent bilateral bone biopsy (day +133) and PET/CT scan (day +143) demonstrated that the focal bone marrow hypermetabolism of 18 F-FDG was caused by the asymmetric distribution of the blasts.…”

Diffuse bone marrow uptake related to granulocyte colony-stimulating factor-producing maxillary sinus carcinoma on 4-borono-2-18F-fluoro-L-phenylalanine positron emission tomography/computed tomography

“…[ 5 ] The overall mortality rate of these tumors is 65.5%, and the overall 1-, 2-, and 5-year survival rate is 57.9%, 44.8%, and 17.7%, respectively. [ 8 9 ] This patient was initially diagnosed with maxillary sinusitis.…”

“…reported the first case of a G-CSF-producing tumor and proposed the following criteria for diagnosing G-CSF-producing tumors: (1) leukocytosis with neutrophil predominance; (2) elevated serum and urine G-CSF levels; (3) normalization of WBC count and serum G-CSF level after removal of the tumor; and (4) increased G-CSF in tumor tissues. [ 8 9 ] The gold standard to diagnose of G-CSF-producing tumor in this case was the laboratory finding. It showed elevated WBC and G-CSF levels, which were accompanied by normalization after the CRT and increase at the recurrence.…”