Polineuropatia hipoglicêmica: registro de caso com insulinoma

Freitas, Marcos R. G. De; Chimelli, Leila; Nascimento, Osvaldo J. M.; Barbosa, Gilberto M.

doi:10.1590/S0004-282X1989000200017

Resumos

Os autores registram o caso de jovem do sexo masculino que desenvolveu polineuropatia sensitivo-motora, com grande amiotrofia das mãos, no curso de crises de. hipoglicemia, provocadas por provável insulinoma. A eletroneuromiografia revelou predominância de alterações axonais. O nervo sural, estudado à microscopia óptica e eletrônica, mostrou perda axonal de fibras mielínicas de grande calibre e de fibras amielínicas. Considerações sobre a etiopatogenia desta síndrome são feitas, parecendo ser devida a hipoglicemia e não a excesso de insulina.

A case of a young man who presented symptoms and clinical signs of polineuropathy that occurred in connection with recurrent hypoglycemic episodes is reported. The hypoglycemia was probably caused by a pancreatic islet tumor. There were, symmetric weakness and wasting of hands and feet, absent tendon reflexes and 'glove and stocking' loss of sensation. Electromyographic studies showed denervation potentials with slight reduction of nerve conduction velocities. Sural nerve biopsy studied by optic and electronic microscopy showed axonal degeneration without signs of demyelination or remyellnation. There are only 30 similar cases reported in the literature. According to experimental findings, the authors believe that glucopenia is the mechanism responsible for the development of the neuropathy, and that at present time there is no evidence for a direct insulin effect.

Polineuropatia hipoglicêmica: registro de caso com insulinoma

Hypoglycemic neuropathy: a case report in association with an insulinoma

Marcos R. G. De Freitas^I; Leila Chimelli^II; Osvaldo J. M. Nascimento^III; Gilberto M. Barbosa^IV

^IDisciplinas de Neurologia e Neuropatologia, Departamentos de Medicina Clínica e de Patologia da Faculdade de Medicina da Universidade Federal Fluminense: Professor Adjunto e Chefe do Serviço de Neurologia

^IIDisciplinas de Neurologia e Neuropatologia, Departamentos de Medicina Clínica e de Patologia da Faculdade de Medicina da Universidade Federal Fluminense: Professor Adjunto de Neuropatologia

^IIIDisciplinas de Neurologia e Neuropatologia, Departamentos de Medicina Clínica e de Patologia da Faculdade de Medicina da Universidade Federal Fluminense: Professor Adjunto de Neurologia

^IVDisciplinas de Neurologia e Neuropatologia, Departamentos de Medicina Clínica e de Patologia da Faculdade de Medicina da Universidade Federal Fluminense: Professor Assistente de Endocrinologia

RESUMO

Os autores registram o caso de jovem do sexo masculino que desenvolveu polineuropatia sensitivo-motora, com grande amiotrofia das mãos, no curso de crises de. hipoglicemia, provocadas por provável insulinoma. A eletroneuromiografia revelou predominância de alterações axonais. O nervo sural, estudado à microscopia óptica e eletrônica, mostrou perda axonal de fibras mielínicas de grande calibre e de fibras amielínicas. Considerações sobre a etiopatogenia desta síndrome são feitas, parecendo ser devida a hipoglicemia e não a excesso de insulina.

SUMMARY

A case of a young man who presented symptoms and clinical signs of polineuropathy that occurred in connection with recurrent hypoglycemic episodes is reported. The hypoglycemia was probably caused by a pancreatic islet tumor. There were, symmetric weakness and wasting of hands and feet, absent tendon reflexes and 'glove and stocking' loss of sensation. Electromyographic studies showed denervation potentials with slight reduction of nerve conduction velocities. Sural nerve biopsy studied by optic and electronic microscopy showed axonal degeneration without signs of demyelination or remyellnation. There are only 30 similar cases reported in the literature. According to experimental findings, the authors believe that glucopenia is the mechanism responsible for the development of the neuropathy, and that at present time there is no evidence for a direct insulin effect.

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1. Daggett P, Nabarro J - Neurological aspects of insulinomas. Postgrad Med J 60: 577, 1984.
2. Danta G - Hypoglycemic peripheral neuropathy. Arch Neurol 21: 121, 1969.
3. Harrison MJG - Muscle wasting after prolonged hypoglycaemic coma: case report with electrophysiological data. J Neurol Neurosurg Psychiat 39: 465, 1976.
4. Hypoglycaemic peripheral neuropathy. Lancet 2:1147, 1982, editorial.
5. Jakobsen J, Sidenius P - Hypoglycaemic neuropathy. In Dyck PJ, Thomas PK, Asbury AK, Winegrad A, Porte D Jr - Diabetic neuropathy. Saunders, Philadelphia, 1987, pg 94.
6. Jaspan JB, Wollmann RL, Bernstein L, Rubenstein AH - Hypoglycemic peripheral neuropathy in association with insulinoma: implication of glucopenia rather than hyper-insulinism. Case report and literature review. Medicine 62: 33, 1982.
7. Jayasinghe KSA, Nimalasuriya A, Dharmadasa K - A case of insulinoma with peripheral neuropathy. Postgrad Med J 59: 189, 1983.
8. Jordan WE - Neuritic manifestations in diabetes mellitus. Arch In;t Med 57:307, 1936.
9. Lambert EH, Mulder DW, Bastron JA - Regeneration of peripheral nerves with hyperinsulin neuronopathy. Neurology 10: 851, 1960.
10. Mandelbaum JA, Felten DL, Westfall SG, Newlin GE, Peterson RG - Neuropathic changes associated with insulin treatment of diabetic rats: electron microscopic and morphometric analysis. Brain Res Bull 10: 377, 1983.
11. Martin MM - Diabetic neuropathy: a clinical study of 150 cases. Brain 76: 594, 1953.
12. Moersch FP, Kernohan JW - Hypoglycemia: neurologic and neuropathology studies. Arch Neurol Psychiat 39: 242, 1938.
13. Mulder DW, Bastron JA, Lambert EH - Hyperinsulin neuronopathy. Neurology 6: 627, 1956.
14. Rundles RW - Diabetic neuropathy: general review with report of 125 cases. Medicine 24: 111, 1945.
15. Sharma AK, Duguid IGM, Blanchard DS, Thomas PK - The efect of insulin treatment on myelinated nerve fibre maturation and integrity and on body growth in streptozotocin-diabetic rats. J Neurol Sci 67: 285, 1985.
16. Sidenius P, Jakobsen J - Peripheral neuropathy in rats induced by insulin treatment. Diabetes 32: 383, 1983.
17. Silfverskiold BP - Polyneuritis hypoglycemia: late peripheral paresis after hipogly-caemic attacks in two insulinoma patients. Acta Med Scand 67: 502, 1946.
18. Stern K, Dancey TE, McNaughton FL - Sensory disturbances following insulin treatment of psychoses. J Nerv Ment Dis 95: 183, 1942.
19. Tom MI, Richardson JC - Hypoglycaemia from islet cell tumor of pancreas with amyotrophy and cerebrospinal nerve cell changes. J Neuropath Exp Neurol 10: 57, 1951.
20. Westfall SG, Felten DL, Mandelbaum JA, Moore SA, Peterson RG - Degenerative neuropathy in insulin-treated diabetic rats. J Neurol Sci 61: 93, 1983.
21. Williams CL - Amyotrophy due to hypoglycaemia. Br Med J 1: 707, 1955.
22. Winkelman NW, Moore MT - Neurohistopathologic changes with metrazol and insulin shock therapy. Arch Neurol Psychiat 43: 1108, 1940.
23. Ziegler DK - Minor neurologic signs and symtons following insulin coma therapy. J Nerv Ment Dis 120: 75, 1954.

Datas de Publicação

Publicação nesta coleção
06 Jun 2011
Data do Fascículo
Jun 1989

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

[1] 1. Daggett P, Nabarro J - Neurological aspects of insulinomas. Postgrad Med J 60: 577, 1984.

[2] 2. Danta G - Hypoglycemic peripheral neuropathy. Arch Neurol 21: 121, 1969.

[3] 3. Harrison MJG - Muscle wasting after prolonged hypoglycaemic coma: case report with electrophysiological data. J Neurol Neurosurg Psychiat 39: 465, 1976.

[4] 4. Hypoglycaemic peripheral neuropathy. Lancet 2:1147, 1982, editorial.

[5] 5. Jakobsen J, Sidenius P - Hypoglycaemic neuropathy. In Dyck PJ, Thomas PK, Asbury AK, Winegrad A, Porte D Jr - Diabetic neuropathy. Saunders, Philadelphia, 1987, pg 94.

[6] 6. Jaspan JB, Wollmann RL, Bernstein L, Rubenstein AH - Hypoglycemic peripheral neuropathy in association with insulinoma: implication of glucopenia rather than hyper-insulinism. Case report and literature review. Medicine 62: 33, 1982.

[7] 7. Jayasinghe KSA, Nimalasuriya A, Dharmadasa K - A case of insulinoma with peripheral neuropathy. Postgrad Med J 59: 189, 1983.

[8] 8. Jordan WE - Neuritic manifestations in diabetes mellitus. Arch In;t Med 57:307, 1936.

[9] 9. Lambert EH, Mulder DW, Bastron JA - Regeneration of peripheral nerves with hyperinsulin neuronopathy. Neurology 10: 851, 1960.

[10] 10. Mandelbaum JA, Felten DL, Westfall SG, Newlin GE, Peterson RG - Neuropathic changes associated with insulin treatment of diabetic rats: electron microscopic and morphometric analysis. Brain Res Bull 10: 377, 1983.

[11] 11. Martin MM - Diabetic neuropathy: a clinical study of 150 cases. Brain 76: 594, 1953.

[12] 12. Moersch FP, Kernohan JW - Hypoglycemia: neurologic and neuropathology studies. Arch Neurol Psychiat 39: 242, 1938.

[13] 13. Mulder DW, Bastron JA, Lambert EH - Hyperinsulin neuronopathy. Neurology 6: 627, 1956.

[14] 14. Rundles RW - Diabetic neuropathy: general review with report of 125 cases. Medicine 24: 111, 1945.

[15] 15. Sharma AK, Duguid IGM, Blanchard DS, Thomas PK - The efect of insulin treatment on myelinated nerve fibre maturation and integrity and on body growth in streptozotocin-diabetic rats. J Neurol Sci 67: 285, 1985.

[16] 16. Sidenius P, Jakobsen J - Peripheral neuropathy in rats induced by insulin treatment. Diabetes 32: 383, 1983.

[17] 17. Silfverskiold BP - Polyneuritis hypoglycemia: late peripheral paresis after hipogly-caemic attacks in two insulinoma patients. Acta Med Scand 67: 502, 1946.

[18] 18. Stern K, Dancey TE, McNaughton FL - Sensory disturbances following insulin treatment of psychoses. J Nerv Ment Dis 95: 183, 1942.

[19] 19. Tom MI, Richardson JC - Hypoglycaemia from islet cell tumor of pancreas with amyotrophy and cerebrospinal nerve cell changes. J Neuropath Exp Neurol 10: 57, 1951.

[20] 20. Westfall SG, Felten DL, Mandelbaum JA, Moore SA, Peterson RG - Degenerative neuropathy in insulin-treated diabetic rats. J Neurol Sci 61: 93, 1983.

[21] 21. Williams CL - Amyotrophy due to hypoglycaemia. Br Med J 1: 707, 1955.

[22] 22. Winkelman NW, Moore MT - Neurohistopathologic changes with metrazol and insulin shock therapy. Arch Neurol Psychiat 43: 1108, 1940.

[23] 23. Ziegler DK - Minor neurologic signs and symtons following insulin coma therapy. J Nerv Ment Dis 120: 75, 1954.