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We chose to undertake a national study, looking at all cases of copper deficiency in Scotland over a 5\u2010yr period using information from a national reference laboratory. From 16 identified patients, we determined that 86% had both haematological and neurological features of copper deficiency, while 18% had haematological features only at presentation. Twelve of the sixteen patients had high serum zinc concentrations (&gt;18 \u03bc<jats:italic>m\/L<\/jats:italic>) nine patients were using zinc\u2010containing dental fixatives at time of diagnosis. 94% of patients had haematological features as an initial manifestation of copper deficiency, which included anaemia, thrombocytopenia and neutropenia. Patients who underwent later bone marrow testing had appearances in keeping with refractory cytopenia with multilineage dysplasia, refractory anaemia with excess of blasts, unclassified marrow dysplasia or probable myelodysplasia (<jats:styled-content style=\"fixed-case\">MDS<\/jats:styled-content>). 75% of patients had neurological symptoms or signs, including progressive walking difficulties and paraesthesia, or gait difficulties without sensory signs. Clinical examination was in keeping with spastic paraparesis (either with or without sensory neuropathy). Magnetic resonance imaging (<jats:styled-content style=\"fixed-case\">MRI<\/jats:styled-content>) showed multifocal<jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>2 hyper intense foci in the subcortical white matter, and atrophy of the cerebrum and cerebellum was also seen on computerised tomography (<jats:styled-content style=\"fixed-case\">CT<\/jats:styled-content>).<jats:styled-content style=\"fixed-case\">MRI<\/jats:styled-content>of the spinal cord showed signal change in the dorsal columns in either the cervical or thoracic cord. 93% of cytopenias responded to copper replacement and addressing the original cause of the copper deficiency, but only 25% of patients had improvement in their neurological function, while 33% deteriorated and 42% remained unchanged. Our study demonstrates that copper deficiency is an under\u2010recognised cause of several types of cytopenia, which are reversible but can progress to significant neurological injury if left untreated. We illustrate the importance of identifying these patients early to prevent irreversible neurological injury.<\/jats:p>","DOI":"10.1111\/ejh.12020","type":"journal-article","created":{"date-parts":[[2012,10,3]],"date-time":"2012-10-03T14:07:09Z","timestamp":1349273229000},"page":"1-9","source":"Crossref","is-referenced-by-count":91,"title":["Hypocupremia associated cytopenia and myelopathy: a national retrospective review"],"prefix":"10.1111","volume":"90","author":[{"given":"Alemayehu A.","family":"Gabreyes","sequence":"first","affiliation":[{"name":"Department of Haematology Glasgow Royal Infirmary Glasgow UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hina Naz","family":"Abbasi","sequence":"additional","affiliation":[{"name":"Department of Neurology Institute of Neurological Sciences Glasgow UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kirsten P.","family":"Forbes","sequence":"additional","affiliation":[{"name":"Department of 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