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Chronic Benign Congenital Myopathy: Fingerprint Body Type

Published online by Cambridge University Press:  18 September 2015

A.S. Gordon ,
N.B. Rewcastle ,
J.G. Humphrey  and
B.M. Stewart
A.S. Gordon
Affiliation:
Division of Neuropathology, Department of Pathology, University of Toronto and St. Michael’s Hospital, Toronto
N.B. Rewcastle*
Affiliation:
Division of Neuropathology, Department of Pathology, University of Toronto and St. Michael’s Hospital, Toronto
J.G. Humphrey
Affiliation:
Division of Neuropathology, Department of Pathology, University of Toronto and St. Michael’s Hospital, Toronto
B.M. Stewart
Affiliation:
Division of Neuropathology, Department of Pathology, University of Toronto and St. Michael’s Hospital, Toronto
*
Dept. of Pathology, University of Toronto, 100 College St, Toronto 2, Canada
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Summary

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The term “benign congenital myopathy” describes a group of muscle disorders characterized by proximal or diffuse muscle weakness, a relatively non-progressive course, normal serum muscle enzyme assays and the presence of a distinctive morphological feature. We report here a 55 year old woman, with fingerprint body myopathy who exhibits all of the above features. She has been affected from birth, able to walk since the age of 12, and has not deteriorated in the past thirty years. Muscle biopsy reveals fatty infiltration, numerous small fibers undergoing structural change, good differentiation into type I and II fibers, and excessive intracellular lipid and lipochrome. Only on electron microscopic study is the distinctive feature of numerous subsarcolemmal round to ovoid fingerprint bodies observed. Many fibers also contain large collections of tubular aggregates, filamentous bodies, and autophagic vacuoles.

The fingerprint bodies are similar to the ones described by A. Engel in a 5 year old girl. Thus, a rare opportunity is provided to study an individual who has had this disease for over fifty years.

Type
Research Article
Information
Canadian Journal of Neurological Sciences , Volume 1 , Issue 2 , May 1974 , pp. 106 - 113
Copyright
Copyright © Canadian Neurological Sciences Federation 1974

References

REFERENCES

Bancroft, J.D. (1967). An Introduction to Histochemical Technique. Butterworth and Company, London.Google Scholar
Bergman, R.A.Afifi, A.K.Dunkle, L.M. and John, R.J. (1970). Muscle pathology in hypokalemic periodic paralysis with hyperthyroidism. John Hopkins Medical Journal, 126, 100–118.Google ScholarPubMed
Brooke, M.H. and Engel, W.K. (1969). The histographic analysis of human muscle biopsies with regard to fiber type. I Adult male and female. Neurology (Minneap) 19, 221–233.CrossRefGoogle Scholar
Brooke, M.H. and Neville, H.E. (1971). Reducing body myopathy. A new disease (Abstract). Neurology (Minneap) 21, 412–413.Google Scholar
Cancilla, P.A.Kalyanaraman, K.Verity, M.A.Munsat, T. and Pearson, C.M. (1971). Familial myopathy with probable lysis of myofibrils in type I fibers. Neurology (Minneap) 21, 579–585.CrossRefGoogle ScholarPubMed
Carpenter, S.Karpati, G.Eisen, A.Anderman, F. and Waters, G. (1972). Childhood dermatomyositis and familial collagen disease (Abstract). Neurology (Minneap) 22, 425.Google Scholar
Chui, L.Neustein, H.Munsat, T.L. and Higgins, J. (1973). Tubular aggregates in alcoholic myopathy (Abstract) Neurology (Minneap) 23, 433–434.Google Scholar
Engel, A.G.Angelini, C. and Gomez, M.R. (1972). Fingerprint body myopathy, a newly recognized congenital muscle disease. Mayo Clinic Proceedings 47, 377–388.Google ScholarPubMed
Engel, A.G.Gomez, M.R. and Groover, R.Y. (1971). Multicore disease. A recently recognized congenital myopathy associated with multifocal degeneration of muscle fibers. Mayo Clinic Proceedings 46, 666–681.Google ScholarPubMed
Engel, A.G. and Macdonald, R.A. (1970). Ultrastructural reactions in muscle disease and their light microscopic correlates, in Muscle Disease: Proceedings of an International Congress, Milan, May 1969. pp.71–89, Editors: Walton, J.N.Canal, N. and Scarlato, G.Excerpta Medica, Amsterdam.Google Scholar
Engel, W.K.Bishop, D.W. and Cunningham, G.G. (1970). Tubular aggregates in type II muscle Fibers: Ultrastructural and histochemical correlation. Journal of Ultrastructural Research 31, 507–525.CrossRefGoogle ScholarPubMed
Engel, W.K.Gold, G.N. and Karpati, G. (1968). Type I fiber hypotrophy and central nuclei. A rare congenital muscle abnormality with a possible experimental model. Archives of Neurology (Chicago) 18, 435–444.Google Scholar
Jerusalem, F.Engel, A.G. and Gomez, M.R. (1973). Sarcotubular myopathy: a newly recognized benign congenital familial muscle disease (Abstract), Neurology (Minneap) 23, 408.Google Scholar
Mair, W.G.P. and Tomé, F.M.S. (1972). Atlas of the Ultrastructure of Diseased Human Muscle. Churchill Livingstone, Edinburgh and London.Google Scholar
Morgan-Hughes, J.A.Mair, W.G.P. and Lascelles, P.T. (1970). A disorder of skeletal muscle associated with tubular aggregates. Brain 93, 873–880.CrossRefGoogle ScholarPubMed
Pearse, A.G.E. and Johnson, M. (1970). Histochemistry in the study of normal disease with special reference to myopathy with tubular aggregates, in Muscle Disease: Proceedings of an International Congress, Milan, May 1969, pp.25–32. Editors: Walton, J.N.Canal, N. and Scarlato, G.Excerpta Medica, Amsterdam.Google Scholar
Shafiq, S.A.Gorycki, M.A.Goldstone, L. and Milhorat, A.T. (1966). Fine structure of fiber types in normal human muscle. Anatomical Record 156, 283–301.CrossRefGoogle Scholar
Sher, J.H.Rimalovski, A.B.Athanassiades, T.J. and Aronson, S.M. (1967). Familial centronuclear myopathy: a clinical and pathological study. Neurology (Minneap) 17, 727–742.CrossRefGoogle ScholarPubMed
Shy, G.M.Engel, W.K.Somers, J.E. and Wanko, T. (1963). Nemaline myopathy: a new congenital myopathy. Brain 86, 793–810.CrossRefGoogle ScholarPubMed
Shy, G.M.Gonatas, N.K. and Perez, M. (1966). Two childhood myopathies with abnormal mitochondria. I. Megaconial myopathy. II. Pleoconial myopathy. Brain 89, 133–158.CrossRefGoogle ScholarPubMed
Shy, G.M. and Magee, K.R. (1956). A new congenital non-progressive myopathy. Brain 79, 610–621.CrossRefGoogle Scholar
Spiro, A.J.Shy, G.M. and Gonatas, N.K. (1966). Myotubular myopathy. Persistence of fetal muscle in an adolecerit boy. Archive of Neurology (Chicago) 14, 1–14.Google Scholar
Tice, L.W. and Engel, A.G. (1967). The effect of glucocorticoids on red and white muscles in the rat. American Journal of Pathology 50, 311–334.Google ScholarPubMed
Tomé, F.M.S. and Fardeau, M. (1973). “Fingerprint inclusions”in muscle fibers in dystrophia myotonica. Acta Neuropathologica. (Berlin) 24, 62–67.Google Scholar