Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos

A consecutive series of 21 patients with single limb atrophy (monomelic amyotrophy) is reported. Sixteen had lower limb atrophy and five had upper limb involvement. The median age of the onset was 20 years. Characteristic features were sporadic occurrence, wasting confined to one limb, insidious ons...

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Main Authors: MARCOS R.G. DE FREITAS, OSVALDO J. M. NASCIMENTO
Format: Article
Language:English
Published: Thieme Revinter Publicações 2000-09-01
Series:Arquivos de Neuro-Psiquiatria
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Online Access:http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X2000000500003
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author MARCOS R.G. DE FREITAS
OSVALDO J. M. NASCIMENTO
author_facet MARCOS R.G. DE FREITAS
OSVALDO J. M. NASCIMENTO
author_sort MARCOS R.G. DE FREITAS
collection DOAJ
description A consecutive series of 21 patients with single limb atrophy (monomelic amyotrophy) is reported. Sixteen had lower limb atrophy and five had upper limb involvement. The median age of the onset was 20 years. Characteristic features were sporadic occurrence, wasting confined to one limb, insidious onset with slow progression, stabilizing in 1 to 4 years, and absence of pyramidal signs. All the patients with upper limb involvement were male, however in our cases with lower limb amyotrophy there were no male preponderance. We observed wasting of the entire length of the lower limbs in six patients. There were nine cases with amyotrophy restricted to the leg and one with amyotrophy only in the thigh. In the upper limb in four cases the involvement was distal and in one patient the atrophy was proximal. The electromyographic features were suggestive of anterior horn disease not only in the affected limb but also, in some cases, in clinically uninvolved limb. Cervical or lumbar MRI was normal. MRI of the lower limb disclosed increased signal intensity in the gastrocnemius and soleus muscles in one patient suggesting denervation.<br>Relatamos uma série consecutiva de 21 pacientes com amiotrofia de um só membro, denominada de amiotrofia monomélica. Em 16 casos a atrofia era no membro inferior e em 5 localizava-se no membro superior. Todos eram jovens e a idade média do início foi 20 anos. Os dados mais característicos da doença foram ausência de história familiar, comprometimento de um só membro, início e progressão lenta estabilizando em até 4 anos e ausência de sinais piramidais. Nossos enfermos com amiotrofia de membro superior eram todos do sexo masculino, entretanto naqueles com amiotrofia do membro inferior havia igualdade de sexos. Dos pacientes com atrofia de membro inferior, em 9 a atrofia era restrita a perna, em 6 era em todo o membro e somente um apresentava amiotrofia localizada só na coxa. Naqueles com comprometimento do membro superior a atrofia era distal em 4 e proximal em 1. Os achados na eletromiografia foram compatíveis com acometimento do II neurônio motor. Todos foram submetidos a RM da coluna cervical ou lombar que se mostrou normal. Em um caso realizamos RM da perna acometida que evidenciou sinais hiperintensos nos músculos gastrocnemius e soleus, o que sugere desnervação.
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spelling doaj-art-0bda59cd24eb4fbd9d6f88faed0edca32025-08-20T01:59:30ZengThieme Revinter PublicaçõesArquivos de Neuro-Psiquiatria0004-282X1678-42272000-09-01583B80881310.1590/S0004-282X2000000500003Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casosMARCOS R.G. DE FREITASOSVALDO J. M. NASCIMENTOA consecutive series of 21 patients with single limb atrophy (monomelic amyotrophy) is reported. Sixteen had lower limb atrophy and five had upper limb involvement. The median age of the onset was 20 years. Characteristic features were sporadic occurrence, wasting confined to one limb, insidious onset with slow progression, stabilizing in 1 to 4 years, and absence of pyramidal signs. All the patients with upper limb involvement were male, however in our cases with lower limb amyotrophy there were no male preponderance. We observed wasting of the entire length of the lower limbs in six patients. There were nine cases with amyotrophy restricted to the leg and one with amyotrophy only in the thigh. In the upper limb in four cases the involvement was distal and in one patient the atrophy was proximal. The electromyographic features were suggestive of anterior horn disease not only in the affected limb but also, in some cases, in clinically uninvolved limb. Cervical or lumbar MRI was normal. MRI of the lower limb disclosed increased signal intensity in the gastrocnemius and soleus muscles in one patient suggesting denervation.<br>Relatamos uma série consecutiva de 21 pacientes com amiotrofia de um só membro, denominada de amiotrofia monomélica. Em 16 casos a atrofia era no membro inferior e em 5 localizava-se no membro superior. Todos eram jovens e a idade média do início foi 20 anos. Os dados mais característicos da doença foram ausência de história familiar, comprometimento de um só membro, início e progressão lenta estabilizando em até 4 anos e ausência de sinais piramidais. Nossos enfermos com amiotrofia de membro superior eram todos do sexo masculino, entretanto naqueles com amiotrofia do membro inferior havia igualdade de sexos. Dos pacientes com atrofia de membro inferior, em 9 a atrofia era restrita a perna, em 6 era em todo o membro e somente um apresentava amiotrofia localizada só na coxa. Naqueles com comprometimento do membro superior a atrofia era distal em 4 e proximal em 1. Os achados na eletromiografia foram compatíveis com acometimento do II neurônio motor. Todos foram submetidos a RM da coluna cervical ou lombar que se mostrou normal. Em um caso realizamos RM da perna acometida que evidenciou sinais hiperintensos nos músculos gastrocnemius e soleus, o que sugere desnervação.http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X2000000500003neurônio motor inferioramiotrofia monomélicaamiotrofia espinhallower motor neuronmonomelic amyotrophyspinal muscular atrophy
spellingShingle MARCOS R.G. DE FREITAS
OSVALDO J. M. NASCIMENTO
Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
Arquivos de Neuro-Psiquiatria
neurônio motor inferior
amiotrofia monomélica
amiotrofia espinhal
lower motor neuron
monomelic amyotrophy
spinal muscular atrophy
title Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
title_full Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
title_fullStr Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
title_full_unstemmed Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
title_short Benign monomelic amyotrophy: a study of twenty-one cases Amiotrofia monomélica benigna: estudo de 21 casos
title_sort benign monomelic amyotrophy a study of twenty one cases amiotrofia monomelica benigna estudo de 21 casos
topic neurônio motor inferior
amiotrofia monomélica
amiotrofia espinhal
lower motor neuron
monomelic amyotrophy
spinal muscular atrophy
url http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X2000000500003
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