Case Report: A Metabolic Disorder Presenting as Pediatric Manganism Vanita Sahni,1 Yves Léger,2 Linda Panaro,1 Mark Allen,3 Scott Giffin,4 Diane Fury,5 and Nadine Hamm5 1Canadian Field Epidemiology Program, Ottawa, Ontario, Canada; 2University of Toronto, Community Medicine Program, Toronto, Ontario, Canada; 3New Brunswick Department of Health, Fredricton, New Brunswick, Canada; 4New Brunswick Health Region 2, St John, New Brunswick, Canada; 5New Brunswick Health Region 1, Moncton, New Brunswick, Canada Abstract Context: Manganese is a trace element, essential for physiologic functioning but neurotoxic at high doses. Common exposure sources include dietary intake as well as drinking water in some regions ; toxicity is most often associated with inhalation exposures in occupational settings. In this article we describe the investigation of a pediatric case of manganism using both clinical and environmental assessment methods. Case presentation: A previously healthy 6-year-old child presented with severe Mn neurotoxicity, iron deficiency, and elevated cobalt levels. Immediate and selected extended family members had elevated plasma Mn but remained asymptomatic. An exposure assessment identified seasonal ingestion exposures to Mn at the family's summer cottage ; these were common to the four immediate family members. Well water used for drinking and cooking exceeded recommended guidelines, and foods high in Mn predominated in their diet. No inhalation exposures were identified. Only pica was unique to the patient. Discussion: The combined evidence of the environmental assessment and biomonitoring of blood Mn levels supported a seasonal ingestion exposure source ; this alone was insufficient to explain the toxicity because the patient's 7-year-old sibling was asymptomatic with almost identical exposures (except pica) . A metabolic disorder involving divalent metals (Mn, Fe, and Co) interacting with environmental exposures is the most likely explanation. Relevance to clinical or professional practice: This case report adds to the emerging body of evidence linking neurologic effects to ingestion Mn exposure. Key words: environmental medicine, manganese, manganese poisoning, toxicity, water. Environ Health Perspect 115:1776–1779 (2007) . doi:10.1289/ehp.10421 available via http://dx.doi.org/ [Online 23 August 2007] Address correspondence to V. Sahni, Canadian Field Epidemiology Program, 130 Colonnade Rd., A.L., 6503A, Ottawa, Ontario, Canada K1A 0K9. Telephone: (613) 327-1268. Fax: (613) 952-3196. E-mail: vanitasahni@hotmail.com We thank the following individuals who were of great assistance during the course of the investigation: K. Gordon, F. Leung, P. Brna, V. Price, S. Dyak, D. Pupek, and T. Arsenault. The authors declare they have no competing financial interests. Received 30 April 2007 ; accepted 23 August 2007. The full version of this article is available for free in HTML or PDF formats. |