Late-onset neurological symptoms in thalidomide-exposed subjects: a study of an Australasian cohort
Background and purpose Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature. Methods This study reviewed 16 of the confirmed...
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| Published in | European journal of neurology Vol. 20; no. 3; pp. 509 - 514 |
|---|---|
| Main Authors | , , |
| Format | Journal Article |
| Language | English |
| Published |
England
Blackwell Publishing Ltd
01.03.2013
John Wiley & Sons, Inc |
| Subjects | |
| Online Access | Get full text |
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| Abstract | Background and purpose
Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature.
Methods
This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new‐onset neurological symptoms in the fourth and fifth decades.
Results
Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living.
Conclusions
The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. |
|---|---|
| AbstractList | Background and purpose
Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature.
Methods
This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new‐onset neurological symptoms in the fourth and fifth decades.
Results
Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living.
Conclusions
The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature. This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new-onset neurological symptoms in the fourth and fifth decades. Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living. The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. Background and purpose Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature. Methods This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new-onset neurological symptoms in the fourth and fifth decades. Results Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living. Conclusions The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. [PUBLICATION ABSTRACT] Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature.BACKGROUND AND PURPOSEThalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature.This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new-onset neurological symptoms in the fourth and fifth decades.METHODSThis study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new-onset neurological symptoms in the fourth and fifth decades.Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living.RESULTSClinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living.The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living.CONCLUSIONSThe study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of thalidomide in its teratogenicity is still actively debated in the literature. This study reviewed 16 of the confirmed Australasian victims of in utero exposure to thalidomide who now presented with new-onset neurological symptoms in the fourth and fifth decades. Clinical neurological examination and neurophysiological investigations revealed that new symptoms were due in part to compressive neuropathies, often exacerbated by the adaptations made to accommodate the disability and poor mobility arising from the limb deformities. Other subjects were found to have musculoskeletal symptoms due to compensatory postures employed to perform tasks of daily living. The study provides no evidence of ongoing loss of neurons or late reactivated neural degeneration and no evidence of a generalized peripheral neuropathy. Rather, the development of new symptoms in subjects can be explained by compressive neuropathies and compensatory postures employed to perform tasks of daily living. |
| Author | Jankelowitz, S. K. Burke, D. Spies, J. M. |
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| Cites_doi | 10.1136/bmj.2.5296.16 10.1054/jhsb.2001.0653 10.1002/(SICI)1096-9926(199612)54:6<273::AID-TERA1>3.0.CO;2-# 10.1097/ICU.0b013e3283499f24 10.1002/mus.880110808 10.1016/j.jhsb.2006.01.001 10.1111/j.1755-3768.1993.tb04997.x 10.1002/bdra.20384 10.5694/j.1326-5377.1963.tb17994.x 10.1016/j.rehab.2009.10.002 10.1080/00039896.1962.10663250 10.1002/tera.1420380303 10.1097/00042752-199601000-00007 |
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| References | Kimura H, Ikuta Y, Ishida O. Carpal tunnel syndrome in radial dysplasia. J Hand Surg [Br] 2001; 26: 533-536. Laffont I, Julia M, Tiffreau V, Yelnik A, Herisson C, Pelissier J. Aging and sequelae of poliomyelitis. Ann Phys Rehabil Med 2010; 53: 24-33. Schuler-Faccini L, Soares RC, de Sousa AC, et al. New cases of thalidomide embryopathy in Brazil. Birth Def Res Part A Clin Mol Terat 2007; 79: 671-672. Miller MT, Strömland K, Ventura L. Congenital aberrant tearing: a re-look. Trans Am Ophthalmol Soc 2008; 106: 100-115. McBride WG. The teratogenic action of drugs. Med J Aust 1963; 2: 689-692. Impink BG, Collinger JL, Boninger ML. The effect of symptoms of carpal tunnel syndrome on ultrasonographic median nerve measures before and after wheelchair propulsion. Postgrad Med Rev 2011; 3: 803-810. Lenz W, Knapp K. Thalidomide embryopathy. Arch Environ Health 1962; 5: 100-105. Miller MT. Thalidomide embryopathy: a model for the study of congenital incomitant horizontal strabismus. Trans Am Ophthalmol Soc 1991; 89: 623-674. Leck IM, Millar EL. Incidence of malformations since the introduction of thalidomide. Br Med J 1962; 2: 16-20. Castilla EE, Ashton-Prolla P, Barreda-Mejia E, et al. Thalidomide, a current teratogen in South America. Teratology 1996; 54: 273-277. Oshima Y, Okutsu I, Hamanaka I, Motomura T. Carpal tunnel syndrome accompanying radial dysplasia due to thalidomide embryopathy. J Hand Surg [Br] 2006; 31: 342-344. Strömland K, Miller MT. Thalidomide embryopathy: revisited 27 years later. Acta Ophthalmol (Copenh) 1993; 71: 238-245. Soliven B, Meer J, Uncini A, Petajan J, Lovelace R. Physiologic and anatomic basis for contralateral R1 in blink reflex. Muscle Nerve 1988; 11: 848-851. Miller MT, Strömland KK. What can we learn from the thalidomide experience: an ophthalmologic perspective. Curr Opin Ophthalmol 2011; 22: 356-364. Jackson DL, Hynninen BC, Caborn DN, McLean J. Electrodiagnostic study of carpal tunnel syndrome in wheelchair basketball players. Clin J Sport Med 1996; 6: 27-31. Lenz WA. A short history of thalidomide embryopathy. Teratology 1988; 38: 203-215. 2010; 53 2006; 31 1991; 89 1962; 5 1993; 71 1963; 2 1988; 38 1962; 2 1988; 11 2011; 22 2008; 106 2001; 26 2011; 3 1996; 54 2007; 79 1996; 6 e_1_2_7_6_1 e_1_2_7_5_1 e_1_2_7_4_1 e_1_2_7_3_1 e_1_2_7_9_1 e_1_2_7_8_1 e_1_2_7_7_1 e_1_2_7_17_1 e_1_2_7_2_1 e_1_2_7_15_1 e_1_2_7_14_1 e_1_2_7_11_1 e_1_2_7_10_1 Impink BG (e_1_2_7_12_1) 2011; 3 Miller MT (e_1_2_7_13_1) 1991; 89 Miller MT (e_1_2_7_16_1) 2008; 106 |
| References_xml | – reference: Miller MT. Thalidomide embryopathy: a model for the study of congenital incomitant horizontal strabismus. Trans Am Ophthalmol Soc 1991; 89: 623-674. – reference: Soliven B, Meer J, Uncini A, Petajan J, Lovelace R. Physiologic and anatomic basis for contralateral R1 in blink reflex. Muscle Nerve 1988; 11: 848-851. – reference: Leck IM, Millar EL. Incidence of malformations since the introduction of thalidomide. Br Med J 1962; 2: 16-20. – reference: Impink BG, Collinger JL, Boninger ML. The effect of symptoms of carpal tunnel syndrome on ultrasonographic median nerve measures before and after wheelchair propulsion. Postgrad Med Rev 2011; 3: 803-810. – reference: Strömland K, Miller MT. Thalidomide embryopathy: revisited 27 years later. Acta Ophthalmol (Copenh) 1993; 71: 238-245. – reference: Miller MT, Strömland K, Ventura L. Congenital aberrant tearing: a re-look. Trans Am Ophthalmol Soc 2008; 106: 100-115. – reference: Lenz WA. A short history of thalidomide embryopathy. Teratology 1988; 38: 203-215. – reference: McBride WG. The teratogenic action of drugs. Med J Aust 1963; 2: 689-692. – reference: Laffont I, Julia M, Tiffreau V, Yelnik A, Herisson C, Pelissier J. Aging and sequelae of poliomyelitis. Ann Phys Rehabil Med 2010; 53: 24-33. – reference: Kimura H, Ikuta Y, Ishida O. Carpal tunnel syndrome in radial dysplasia. J Hand Surg [Br] 2001; 26: 533-536. – reference: Oshima Y, Okutsu I, Hamanaka I, Motomura T. Carpal tunnel syndrome accompanying radial dysplasia due to thalidomide embryopathy. J Hand Surg [Br] 2006; 31: 342-344. – reference: Jackson DL, Hynninen BC, Caborn DN, McLean J. Electrodiagnostic study of carpal tunnel syndrome in wheelchair basketball players. Clin J Sport Med 1996; 6: 27-31. – reference: Lenz W, Knapp K. Thalidomide embryopathy. Arch Environ Health 1962; 5: 100-105. – reference: Miller MT, Strömland KK. What can we learn from the thalidomide experience: an ophthalmologic perspective. Curr Opin Ophthalmol 2011; 22: 356-364. – reference: Castilla EE, Ashton-Prolla P, Barreda-Mejia E, et al. Thalidomide, a current teratogen in South America. Teratology 1996; 54: 273-277. – reference: Schuler-Faccini L, Soares RC, de Sousa AC, et al. New cases of thalidomide embryopathy in Brazil. Birth Def Res Part A Clin Mol Terat 2007; 79: 671-672. – volume: 2 start-page: 16 year: 1962 end-page: 20 article-title: Incidence of malformations since the introduction of thalidomide publication-title: Br Med J – volume: 22 start-page: 356 year: 2011 end-page: 364 article-title: What can we learn from the thalidomide experience: an ophthalmologic perspective publication-title: Curr Opin Ophthalmol – volume: 11 start-page: 848 year: 1988 end-page: 851 article-title: Physiologic and anatomic basis for contralateral R1 in blink reflex publication-title: Muscle Nerve – volume: 3 start-page: 803 year: 2011 end-page: 810 article-title: The effect of symptoms of carpal tunnel syndrome on ultrasonographic median nerve measures before and after wheelchair propulsion publication-title: Postgrad Med Rev – volume: 2 start-page: 689 year: 1963 end-page: 692 article-title: The teratogenic action of drugs publication-title: Med J Aust – volume: 89 start-page: 623 year: 1991 end-page: 674 article-title: Thalidomide embryopathy: a model for the study of congenital incomitant horizontal strabismus publication-title: Trans Am Ophthalmol Soc – volume: 38 start-page: 203 year: 1988 end-page: 215 article-title: A short history of thalidomide embryopathy publication-title: Teratology – volume: 31 start-page: 342 year: 2006 end-page: 344 article-title: Carpal tunnel syndrome accompanying radial dysplasia due to thalidomide embryopathy publication-title: J Hand Surg [Br] – volume: 53 start-page: 24 year: 2010 end-page: 33 article-title: Aging and sequelae of poliomyelitis publication-title: Ann Phys Rehabil Med – volume: 6 start-page: 27 year: 1996 end-page: 31 article-title: Electrodiagnostic study of carpal tunnel syndrome in wheelchair basketball players publication-title: Clin J Sport Med – volume: 106 start-page: 100 year: 2008 end-page: 115 article-title: Congenital aberrant tearing: a re‐look publication-title: Trans Am Ophthalmol Soc – volume: 26 start-page: 533 year: 2001 end-page: 536 article-title: Carpal tunnel syndrome in radial dysplasia publication-title: J Hand Surg [Br] – volume: 5 start-page: 100 year: 1962 end-page: 105 article-title: Thalidomide embryopathy publication-title: Arch Environ Health – volume: 71 start-page: 238 year: 1993 end-page: 245 article-title: Thalidomide embryopathy: revisited 27 years later publication-title: Acta Ophthalmol (Copenh) – volume: 54 start-page: 273 year: 1996 end-page: 277 article-title: Thalidomide, a current teratogen in South America publication-title: Teratology – volume: 79 start-page: 671 year: 2007 end-page: 672 article-title: New cases of thalidomide embryopathy in Brazil publication-title: Birth Def Res Part A Clin Mol Terat – ident: e_1_2_7_4_1 doi: 10.1136/bmj.2.5296.16 – ident: e_1_2_7_9_1 doi: 10.1054/jhsb.2001.0653 – ident: e_1_2_7_6_1 doi: 10.1002/(SICI)1096-9926(199612)54:6<273::AID-TERA1>3.0.CO;2-# – ident: e_1_2_7_15_1 doi: 10.1097/ICU.0b013e3283499f24 – volume: 89 start-page: 623 year: 1991 ident: e_1_2_7_13_1 article-title: Thalidomide embryopathy: a model for the study of congenital incomitant horizontal strabismus publication-title: Trans Am Ophthalmol Soc – ident: e_1_2_7_17_1 doi: 10.1002/mus.880110808 – ident: e_1_2_7_10_1 doi: 10.1016/j.jhsb.2006.01.001 – ident: e_1_2_7_14_1 doi: 10.1111/j.1755-3768.1993.tb04997.x – ident: e_1_2_7_7_1 doi: 10.1002/bdra.20384 – ident: e_1_2_7_2_1 doi: 10.5694/j.1326-5377.1963.tb17994.x – ident: e_1_2_7_8_1 doi: 10.1016/j.rehab.2009.10.002 – ident: e_1_2_7_3_1 doi: 10.1080/00039896.1962.10663250 – ident: e_1_2_7_5_1 doi: 10.1002/tera.1420380303 – volume: 3 start-page: 803 year: 2011 ident: e_1_2_7_12_1 article-title: The effect of symptoms of carpal tunnel syndrome on ultrasonographic median nerve measures before and after wheelchair propulsion publication-title: Postgrad Med Rev – ident: e_1_2_7_11_1 doi: 10.1097/00042752-199601000-00007 – volume: 106 start-page: 100 year: 2008 ident: e_1_2_7_16_1 article-title: Congenital aberrant tearing: a re‐look publication-title: Trans Am Ophthalmol Soc |
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Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact... Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact mechanism of action of... Background and purpose Thalidomide was marketed for the treatment of morning sickness and resulted in foetal death and physical deformities. The exact... |
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| Title | Late-onset neurological symptoms in thalidomide-exposed subjects: a study of an Australasian cohort |
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