Summary
A necrotizing, nonprogressive myopathy with unusual paracrystalline inclusion bodies is described in a patient who underwent long-term treatment with megadoses of vitamin E. The clinical course and morphological findings suggest a close relationship to the administration of the vitamin. The theoretical pathogenesis of muscle damage and the possible origin of paracrystalline inclusion bodies are discussed.
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References
Balisteri WF, Farrell MK, Bove KE (1986) Lessons from the E-ferol tragedy. Pediatrics 78:503–506
Bardosi A, Zobel HJ (1985) A new clamp for muscle and nerve biopsy. Muscle Nerve 8:725
Bardosi A, Creutzfeld W, DiMauro S, Felgenhauer K, Friede RL, Goebel HH, Kohlschütter A, Mayer G, Rahlf G, Servidei S, Van Lessen G, Wetterling T (1987) Myoneuro-gastrointestinal encephalopathy (MNGIE syndrome) due to partial deficiency of cytochrome-c-oxidase. A new mitochondrial multisystem disorder. Acta Neuropathol (Berl) 74:248–258
Bieri JG, Corash L, Hubbard VS (1983) Medical uses of vitamin E. N Engl J Med 308:1063–1071
Boxer LA, Oliver JM, Spielberg SP, Allen JM, Schulman JD (1979) Protection of granulocytes by vitamin E in glutathione synthetase deficiency. N Engl J Med 301:901–905
Briggs M (1974) Vitamin E supplements and fatigue. N Engl J Med 290:579–580
Burck U, Goebel HH, Kuhlendahl HD, Meier C, Goebel KM (1981) Neuromyopathy and vitamin E deficiency in man. Neuropediatrics 12:267–278
Cohen HM (1973) To the Editor: Effects of vitamin E: good and bad. N Engl J Med 289:980
Corash L, Spielberg S, Bartsocas C, Boxer L, Steinherz R, Sheetz M, Egan M, Schlessleman J, Schulman JD (1980) Reduced chronic hemolysis during high-dose vitamin E administration in mediterranean-type glucose-6-phosphate dehydrogenase deficiency. N Engl J Med 303:416–420
Dickson J, Hopkins DL, Doncon GH (1986) Muscle damage associated with injections of vitamin E in sheep. Aust Vet J 63:231–233
Divakaran P, Wiggins RC (1987) Tocopherol in brain metabolism and disease: a review. Metab Brain Dis 2:1–16
Ehrenkranz RA, Bonta BW, Ablow RC, Warshaw JB (1978) Amelioration of bronchopulmonary dysplasia after vitamin E administration. A preliminary report. N Engl J Med 299:564–569
Finer NN, Peters KL, Hayek Z, Merkel CL (1984) Vitamin E and necrotizing enterocolitis. Pediatrics 73:387–393
Fragata M, Bellemare F (1980) Model of singlet oxygen scavenging by α-tocopherol in biomembranes. Chem Phys Lipids 27:93–99
Graber B, Guggenheim MA, Ringel PC, Neville HE (1980) Neuromuscular disease related to chronic vitamin E deficiency. Ann Neurol 8:217
Herbert V (1977) Vitamin E report. Nutr Rev 35:158
Hittner HM, Godio LB, Rudolph AJ, Adams JM, Garcia-Prats JA, Friedman Z, Kautz JA, Monaco WA (1981) Retrolental fibroplasia: efficacy of vitamin E in a double-blind clinical study of preterm infants. N Engl J Med 305:1365–1371
Ishikawa H (1986) Formation of elaborate networks of T-system tubules in cultured skeletal muscle with special reference to the T-system formation. J Cell Biol 38:51–66
Johnson L, Bowen FW Jr, Abbasi S, Herrmann N, Weston M, Sacks L, Porat R, Stahl G, Peckham G, Delivoria-Papadopoulos M, Quinn G, Schaffer D (1985) Relationship of prolonged pharmacologic serum levels of vitamin E incidence of sepsis and necrotizing enterocolitis in infants with birth weight 1,500 grams or less. Pediatrics 75:619–638
Kligman AM (1982) Vitamin E toxicity. Arch Dermatol 118:289
Lorch V, Murphy D, Hoersten LR, Harris E, Fitzgerald J, Sinha SN (1985) Unusual syndrome among premature infants associated with a new intravenous vitamin E product. Pediatrics 75:598–602
Lucy JA (1972) Functional and structural aspects of biological membranes: a suggested structural role for vitamin E in the control of membrane permeability and stability. Ann NY Acad Sci 203:4–11
Murphy BF (1974) Hypervitaminosis E. JAMA 227:1381
Natta CL, Machlin LJ, Brin M (1980) A decrease in irreversibly sickled erythrocytes in sickle cell anemia patients given vitamin E. Am J Clin Nutr 33:968–971
Neville HE, Ringel SP, Guggenheim MA, Wehling C (1980) Histochemical and ultrastructural abnormalities of muscle in a patient with chronic vitamin E deficiency. Neurology 30:404
Neville HE, Ringel SP, Guggenheim MA, Wehling CA, Starcevich JM (1983) Ultrastructural and histochemical abnormalities of skeletal muscle in patients with chronic vitamin E deficiency. Neurology 33:483–488
Palmucci L, Anzil AP, Luh S (1983) Crystalline aggregates of protein-glycogen complexes (alias ‘virus-like particles’) in skeletal muscle: report of a case and review of the literature. Neuropathol Appl Neurobiol 9:61–71
Phelps DL (1984) E-ferol: what happened and what now? Pediatrics 74:1114–1116
Prasad JS (1980) Effect of vitamin E supplementation on leukocyte function. Am J Clin Nutr 33:606–608
Rachmilewitz EA, Shifter A, Kahane I (1979) Vitamin E deficiency in β-thalassemia major: changes in hematological and biochemical parameters after a therapeutic trial with α-tocopherol. Am J Clin Nutr 32:1850–1858
Roberts HJ (1978) Vitamin E and thrombophlebitis. Lancet I:49
Roberts HJ (1979) Thrombophlebitis associated with vitamin E therapy: with a commentary on other medical side effects. Agniology 30:169–176
Robert HJ (1981) Perspective on vitamin E as therapy. JAMA 246:129–131
Schwarz K (1972) The cellular mechanism of vitamin E action: direct and indirect effects of α-tocopherol on mitochondrial respiration. Ann NY Acad Sci 203:45–52
Takenaga K, Honma Y, Hozumi M (1981) Inhibition of differentiation of mouse myeloid leukemia cells by phenolic antioxidants and α-tocopherol. Gann 72:104–112
Tappel AL (1972) Vitamin E and free radical peroxidation of lipids. Ann NY Acad Sci 203:12–28
Umetsu DT, Couture P, Winter HS, Kagen BM, Bresnan MJ, Lux SE (1980) Degenerative neurological disease in patients with acquired vitamin E deficiency. Pediatr Res 14:512
Wahlquist ML, Briggs DR, Jones GP (1982) Vitamins E, C and the B complex. Aust Fam Physician 11:270–278
Werlin SL, Harb JM, Swick H, Blank E (1983) Neuromuscular dysfunction and ultrastructural pathology in children with chronic cholestasis and vitamin E deficiency. Ann Neurol 13:291–296
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This study was supported by a grant from the Deutsche Forschungsgemeinschaft (Ba 916)
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Bardosi, A., Dickmann, U. Necrotizing myopathy with paracrystalline inclusion bodies in hypervitaminosis E. Acta Neuropathol 75, 166–172 (1987). https://doi.org/10.1007/BF00687078
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DOI: https://doi.org/10.1007/BF00687078