Introduction: Autoimmune thyroid disease is frequently associated with other immune-mediated disorders; however, clinically significant pancytopenia is rare. In patients with Graves’ disease receiving antithyroid therapy, leukopenia raises concern for drug-induced agranulocytosis, a rare but potentially life-threatening complication characterized by severe neutropenia requiring immediate drug withdrawal. The coexistence of hemolytic anemia and thrombocytopenia may instead suggest Evans syndrome, defined by autoimmune hemolytic anemia with immune thrombocytopenia, with or without neutropenia. Importantly, uncontrolled thyrotoxicosis itself may cause immunemediated cytopenias, creating a diagnostic challenge. Case: We report a 55-year-old female with thyroid receptor antibody-positive Graves’ disease who presented with jaundice and pancytopenia while receiving carbimazole therapy. Laboratory evaluation demonstrated anemia with reticulocytosis and a positive direct antiglobulin test, thrombocytopenia and leukopenia. Complement testing revealed reduced C3 with normal C4, consistent with immune-mediated hemolysis. Peripheral blood film showed no blast cells or marrow infiltration, and autoimmune screening, including antinuclear antibodies and anti–double stranded DNA, was negative. The coexistence of Coombs-positive hemolysis and thrombocytopenia initially raised suspicion for Evans syndrome, while leukopenia during carbimazole therapy prompted concern for drug-induced agranulocytosis. However, neutropenia was not severe, and the absence of marrow infiltration or systemic autoimmune disease made alternative causes of pancytopenia less likely. Importantly, blood counts progressively improved following the optimization of thyroid control despite continuation of carbimazole at a reduced dose, without the use of immunosuppressive therapy. This clinical course supported the interpretation of thyrotoxicosis-associated immune cytopenia rather than primary Evans syndrome or carbimazole-induced agranulocytosis. Conclusion This case highlights thyrotoxicosis-associated immune cytopenia as an important mimic of Evans syndrome and carbimazole-related hematological toxicity. Recognizing this entity is essential to avoid unnecessary discontinuation of antithyroid therapy or inappropriate immunosuppressive treatment.
Evans Syndrome ; Carbimazole ; Pancytopenia ; Agranulocytosis ; Graves Disease

Introduction: Graves’ disease is associated with various hematological abnormalities, including anemia, leucopenia, and thrombocytopenia. However, pancytopenia involving all three cell lines is a rare and often under-recognized manifestation of thyrotoxicosis. We present a case of newly diagnosed Graves’ disease complicated by pancytopenia, in which cell counts normalized rapidly following carbimazole initiation. Case: A 51-year-old female with underlying type 2 diabetes mellitus, hypertension, and dyslipidemia presented with dysphagia for 3 months, significant weight loss (from 95 to 75 kg over 6 months), and a 1-week history of fever, palpitations, tremors, orthopnea, and bilateral lower limb swelling. On examination, she was febrile (38.2°C) with bibasal crepitations, pitting edema up to the midshins, bilateral hand tremors, and a multinodular neck mass moving with deglutition. Investigations confirmed thyrotoxicosis (thyroid-stimulating hormone [TSH] 0.01 mIU/L, free T4 130.1 pmol/L, T3 >30.8 pmol/L) with positive autoantibodies (thyroid receptor antibody 31.9 IU/L, anti-thyroid peroxidase 195 IU/mL). Full blood count showed pancytopenia: white cell count 2.73 × 10⁹/L, hemoglobin 10.3 g/dL, and platelets 108 × 10⁹/L. Peripheral blood film suggested normocytic normochromic anemia with leucopenia and thrombocytopenia. Chest radiography showed cardiomegaly with fluid overload, and echocardiography revealed an ejection fraction of 77%. She was treated for impending thyroid storm secondary to pneumonia with Lugol’s iodine, hydrocortisone, propylthiouracil, and intravenous antibiotics. She was discharged on day 3 with a transition to carbimazole. At outpatient follow-up approximately 9 days later, thyroid function had improved significantly (free thyroxine 4 reduced from 130.1 to 29.34 pmol/L with suppressed TSH), and repeat full blood count demonstrated complete normalization of all three cell lines. Conclusion This case illustrates that pancytopenia can be a direct consequence of severe thyrotoxicosis and may reverse completely with effective antithyroid therapy. The temporal relationship between biochemical improvement and hematological recovery supports a causal link. Clinicians should be aware of this rare association to avoid misdiagnosis and unnecessary invasive investigations.
Pancytopenia ; Graves Disease ; Thyrotoxicosis

Child ; Humans ; Intracellular Signaling Peptides and Proteins ; Mutation ; Pancytopenia

Anemia, Aplastic ; Bone Marrow Diseases ; Bone Marrow Failure Disorders ; Humans ; Pancytopenia

Metastasis of rhabdomysarcoma to the breast is a very rare manifestation in adult males. Herein, we report a case of metastasis from embryonal rhabdomyosarcoma in the left hypothenar muscle that presented as a breast mass in a 38-year-old man, who four months later expired because of multiple bone metastases related to pancytopenia. We describe the various imaging findings, including mammograms, ultrasonography, computerized tomography (CT), positron emission tomography-computed tomography (PET-CT), and magnetic resonance imaging (MRI) of this rare disease. The various imaging findings of this lesion could be helpful for future diagnosis of male breast lesions.
Adult ; Breast ; Diagnosis ; Electrons ; Humans ; Magnetic Resonance Imaging ; Male ; Neoplasm Metastasis ; Pancytopenia ; Rare Diseases ; Rhabdomyosarcoma ; Rhabdomyosarcoma, Embryonal ; Ultrasonography

PURPOSE: The major side effects of treatment with oxcarbazepine (OXC) are skin rash and hyponatremia. Hematologic side effects are reported rarely. The aim of this study was to investigate the rate and types of the hematologic side effects of OXC. METHODS: The medical records of 184 patients diagnosed with epilepsy or movement disorder and on OXC monotherapy, at the Department of Pediatrics of Inje University Sanggye Paik Hospital from July 2001 to July 2018, were retrospectively reviewed. RESULTS: Of the 184 patients, 10 (5.4%) developed leukopenia in addition to pancytopenia and 2 (1.0%) developed pancytopenia. Leukopenia developed in 11 days to 14 years after OXC administration and was more frequent in males than in females (male vs. female, 9 vs. 1; Fisher exact test, P<0.05). Of the eight patients with leukopenia alone, 7 continued OXC treatment; 6 improved without intervention; 1 was lost to follow-up; and 1 received a reduced OXC dose, who improved after intervention. Pancytopenia developed within 2 months of initiation of OXC treatment. Both patients initially continued OXC. One improved within 1 month and continued treatment with OXC, but the other showed progression of the side effect, leading to the discontinuation of OXC and subsequent improvement within 1 month. There were no significant differences in the ages of the patients, OXC dose, and duration of OXC treatment between patients with and without these side effects of OXC (P >0.05, t-test). CONCLUSION: OXC-induced leukopenia is not rare and may result in pancytopenia. Patients being treated with OXC should be regularly monitored for abnormal complete blood count profiles.
Blood Cell Count ; Epilepsy ; Exanthema ; Female ; Humans ; Hyponatremia ; Leukopenia ; Lost to Follow-Up ; Male ; Medical Records ; Movement Disorders ; Pancytopenia ; Pediatrics ; Retrospective Studies

Bone marrow metastasis of colon cancer is rare. Here, we report a 56-year-old female patient who presented with pancytopenia. She was diagnosed with colon cancer accompanied by lung and axial skeleton metastasis. The bone marrow study showed metastatic carcinoma. Immunohistochemical (IHC) staining with anti-cytokeratin 7 (CK7) and anti-cytokeratin 20 (CK20) antibodies showed that the bone marrow samples were negative for CK7 and positive for CK20, consistent with metastatic colon cancer. To the best of our knowledge, there has been only one other reported case of bone marrow metastasis of colon cancer as the primary diagnosis in an adult patient in Korea. Bone and bone marrow metastases of colon cancer are regarded as uncommon. However, for proper management, bone marrows should be promptly examined in patients with solid tumors when unexplained cytopenia is noted, even if the origin of the tumor is known to be rarely metastatic to bone marrow. In addition, the use of cytokeratin IHC staining is helpful for determining the origin of metastatic carcinoma.
Adult ; Antibodies ; Bone Marrow ; Colon ; Colonic Neoplasms ; Diagnosis ; Female ; Humans ; Immunohistochemistry ; Keratins ; Korea ; Lung ; Middle Aged ; Neoplasm Metastasis ; Pancytopenia ; Skeleton

A 22-year old female patient with systemic lupus erythematosus presenting microangiopathic hemolytic anemia was treated with therapeutic plasma exchange 23 times. The patient's condition and laboratory findings (aspartate aminotransferase, alanine aminotransferase, ferritin, total bilirubin, and lactate dehydrogenase) did not improve despite the initial 18 therapeutic plasma exchange treatments. Thrombotic thrombocytopenic purpura was ruled out due to normal ADAMTS-13 activity test result; hemophagocytic lymphohistiocytosis was diagnosed based on fever, splenomegaly, pancytopenia, hypertriglyceridemia, hyperferritinemia, and hemophagocytosis in bone marrow aspiration. The patient's condition improved rapidly upon treatment with a combination of immunosuppressants and cytotoxic agents, and more therapeutic plasma exchanges were performed five consecutive times with prolonged intervals in between. We observed that therapeutic plasma exchange treatment alone was not effective enough to treat hemophagocytic lymphohistiocytosis, unlike thrombotic thrombocytopenic purpura. Therefore, it is necessary to determine and start drug administration promptly in the treatment of hemophagocytic lymphohistiocytosis with thrombotic microangiopathy.
Alanine Transaminase ; Anemia, Hemolytic ; Bilirubin ; Bone Marrow ; Cytotoxins ; Female ; Ferritins ; Fever ; Humans ; Hypertriglyceridemia ; Immunosuppressive Agents ; Lactic Acid ; Lupus Erythematosus, Systemic ; Lymphohistiocytosis, Hemophagocytic ; Pancytopenia ; Plasma Exchange ; Plasma ; Purpura, Thrombotic Thrombocytopenic ; Splenomegaly ; Thrombotic Microangiopathies

OBJECTIVETo investigate the potential efficacy of panaxadiol saponins component (PDS-C), a biologically active fraction isolated from total ginsenosides, to reverse chemotherapy-induced myelosuppression and pancytopenia caused by cyclophamide (CTX).

METHODSMice with myelosuppression induced by CTX were treated with PDS-C at a low- (20 mg/kg), moderate- (40 mg/kg), or high-dose (80 mg/kg) for 7 consecutive days. The level of peripheral white blood cell (WBC), neutrophil (NEU) and platelet (PLT) were measured, the histopathology and colony formation were observed, the protein kinase and transcription factors in hematopoietic cells were determined by immunohistochemical staining and Western blot.

RESULTSIn response to PDS-C therapy, the peripheral WBC, NEU and PLT counts of CTX-induced myelosuppressed mice were significantly increased in a dose-dependent manner. Similarly, bone marrow histopathology examination showed reversal of CTX-induced myelosuppression with increase in overall bone marrow cellularity and the number of hematopoietic cells (P<0.01). PDS-C also promoted proliferation of granulocytic and megakaryocyte progenitor cells in CTX-treated mice, as evidenced by significantly increase in colony formation units-granulocytes/monocytes and -megakaryocytes (P<0.01). The enhancement of hematopoiesis by PDS-C appears to be mediated by an intracellular signaling pathway, this was evidenced by the up-regulation of phosphorylated mitogen-activated protein kinase (p-MEK) and extracellular signal-regulated kinases (p-ERK), and receptor tyrosine kinase (C-kit) and globin transcription factor 1 (GATA-1) in hematopoietic cells of CTX-treated mice (P<0.05).

CONCLUSIONSPDS-C possesses hematopoietic growth factor-like activities that promote proliferation and also possibly differentiation of hematopoietic progenitor cells in myelosuppressed mice, probably mediated by a mechanism involving MEK and ERK protein kinases, and C-kit and GATA-1 transcription factors. PDS-C may potentially be a novel treatment of myelosuppression and pancytopenia caused by chemotherapy.

Animals ; Antineoplastic Agents ; adverse effects ; Cell Proliferation ; drug effects ; Cyclophosphamide ; adverse effects ; Extracellular Signal-Regulated MAP Kinases ; metabolism ; GATA1 Transcription Factor ; metabolism ; Ginsenosides ; pharmacology ; therapeutic use ; Hematopoiesis ; drug effects ; Mice ; Mitogen-Activated Protein Kinase Kinases ; metabolism ; Myeloid Cells ; drug effects ; pathology ; Panax ; chemistry ; Pancytopenia ; chemically induced ; drug therapy ; pathology ; Phosphorylation ; drug effects ; Proto-Oncogene Proteins c-kit ; metabolism ; Saponins ; pharmacology ; Up-Regulation ; drug effects

Is it necessary to intake anthelmintics every year in Korea? To answer to this question, the recent nation-wide egg positive rate of the intestinal nematodes in Korea was presented. The anthelminthics which are purchasable without physician's prescription were also introduced with their pharmacological reaction and indication. The egg positive rate of Ascaris lumbricoides in 2012 was 0.025%. Those of Trichuris trichiura and Enterobius vermicularis were 0.27% and 0.004%, respectively. In 2018, purchasable anthelmintics without physician's prescription in Korea were albendazole and flubendazole only. Those two anthelmintics were derivatives of benzimidazole that may cause some side effects such as hepatitis, increase of hepatic enzymes, granulocytopenia, or pancytopenia. These anthelmintics showed excellent effect to ascariasis; while, they are not sufficient to treat trichuriaiss. For treatment of enterobiasis, repeated taking 3 times with 3 weeks interval and mass treatment of the family of egg positive person are required. In conclusion, it is not necessary to take anthelmintics every year without specific diagnosis because of negligible egg positive rate of intestinal nematodes and complicated therapeutic module for enterobiasis. There was no specific symptom of ascariasis or trchuriasis if worm burden is not high. The common symptoms of enterobiasis were pain or itching at the perianal area, sleep difficulty, or diarrhea. If intestinal nematode infection is suspected, stool examination or perianal swab should be done before prescribing anthelmintics.
Agranulocytosis ; Albendazole ; Anthelmintics ; Ascariasis ; Ascaris lumbricoides ; Diagnosis ; Diarrhea ; Enterobiasis ; Enterobius ; Hepatitis ; Humans ; Korea ; Nematode Infections ; Ovum ; Pancytopenia ; Prescriptions ; Pruritus ; Trichuris