Introduction: hematological disorders are among the so-called unusual manifestations of hypothyroidism, and by far dominated by anemia. Leucocytes abnormalities are less common and often of an autoimmune nature. We report an original case of isolated non-autoimmune neutropenia revealing primary severe hypothyroidism.Case report: A 30-year-old woman was referred by her family doctor for exploration of leucopenia at 3200/mm 3 . Control of the blood count confirmed isolated neutropenia at 600/mm 3 . The other parameters of the total blood count were without abnormalities: hemoglobin at 13g/dl, platelets at 162000/mm 3 , and lymphocytes at 2400/mm 3 . Clinical examination, baseline bioassays, abdominal ultrasonography, thoraco-abdominopelvic CT, and myelogram were without abnormalities. Anti-nuclear, antinative DNA, anti-nucleosome, and anti-neutrophil antibodies were negative.Subsequent investigations concluded to primary hypothyroidism of Hashimoto's thyroiditis: TSH at 182μU/ml, FT4<1 pmol/l, anti-thyroglobulin antibodies at 255 IU/ml, and anti-thyroperoxidase antibodies at 1000 IU/ml. The patient was treated with thyroxine in progressive doses until normalization of TSH with 175μg/d. The evolution was favorable with neutrophils at 2200/mm 3 after one month of treatment.
Conclusion:Isolated and non-autoimmune neutropenia indicative of hypothyroidism is exceptional and unusual representing a real diagnostic challenge for clinicians. It is recommended to screen for thyroid dysfunction in front of any leucopenia that does not prove itself.