Early neurophysiological evolution of chronic inflammatory demyelinating polyneuropathy in a patient with Hashimoto's thyroiditis

Bairactaris, Chrisostomos; Stouraitis, Georgios; Papalias, Elias; Fakas, Nikolaos; Papatheodorou, Georgios; Gourtzelidis, Pavlos

doi:10.1002/mus.21138

Cited by 13 publications

(7 citation statements)

References 23 publications

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“…Interestingly, there was also a report on an adult patient with Hashimoto's thyroiditis who developed isolated peripheral nervous system involvement consisting of chronic inflammatory demyelinating polyneuropathy; in that case neurophysiological tests indicated prolonged sensory and motor terminal latencies, slowing of conduction velocities, conduction block in nerve segments and absent F‐waves. That patient received high‐dose oral prednisolone with an excellent response and 1 month later he was in complete remission . To our knowledge, peripheral nervous involvement as in the present case has not been previously reported in the pediatric literature.…”

Section: Discussionsupporting

confidence: 50%

Pediatric Hashimoto's encephalopathy with peripheral nervous system involvement

Salpietro

Mankad

Polizzi³

et al. 2014

Pediatrics International

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Hashimoto encephalopathy is a syndrome of encephalopathy associated with elevated concentration of circulating serum anti-thyroid antibodies usually responsive to steroid therapy. We report a 13-year-old girl with Hashimoto encephalopathy and peripheral nervous system involvement. The child had experienced high-grade pyrexia, global headache and sleeplessness. After admission she had an ileus with a distended urinary bladder, hallucinations and cognitive impairment. She had reduced deep tendon reflexes and distal sensory deficiency. Anti-thyroglobulin antibodies were raised at 2121 IU/mL (normal, 0-40) and the anti-thyroperoxidase was high at 886 IU/mL (normal, 0-50). Progressive neurological and psychiatric remission was noted after i.v. methylprednisolone. Follow-up magnetic resonance imaging showed complete resolution of the foci of signal abnormality previously yielded. This case report is the first, to the best of our knowledge, to describe peripheral nervous system involvement in a child with a diagnosis of Hashimoto's encephalopathy.

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Section: Discussionsupporting

confidence: 50%

Pediatric Hashimoto's encephalopathy with peripheral nervous system involvement

Salpietro

Mankad

Polizzi³

et al. 2014

Pediatrics International

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“…Therefore, the single parietooccipital lesion and visual-evoked potential findings were evaluated as tumefactive CNS involvement of MADSAM neuropathy, a previously unreported observation. The presence of autoimmune thyroiditis in this patient would be far more than a coincidence because there are reports of autoimmune syndrome complexes involving Hashimoto's thyroiditis, CIDP or CNS demyelination (20). CNS or peripheral demyelination can occur even though thyroid involvement is asymptomatic.…”

Section: Discussionmentioning

confidence: 76%

Tumefactive Brain Demyelination Accompanying MADSAM Neuropathy

Erdener¹,

Temuçin²,

Söylemezoğlu³

et al. 2015

tnd

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Multifocal acquired demyelinating sensory and motor (MADSAM) neuropathy is characterized by asymmetric multifocal motor and sensory loss and conduction blocks in peripheral nerves. Peripheral demyelinating diseases may be accompanied by demyelination in the central nervous system (CNS). In this report, we present a patient with MADSAM who had a solitary tumefactive demyelinating lesion in the brain. Neuroimaging was performed because of a visual field defect revealed a right parietooccipital lesion, which was initially misdiagnosed as a tumor. A pathologic examination showed that it was demyelinating in nature. The patient developed peripheral nervous symptoms two years later and was then diagnosed as having MADSAM. There was a prominent clinical and electrophysiologic response to steroid treatment. Tumefactive brain involvement has not previously been reported for MADSAM neuropathy, although it has been documented in a single case of typical chronic inflammatory demyelinating polyneuropathy (CIDP). CNS involvement should therefore be considered in patients with MADSAM. Keywords: Multifocal acquired demyelinating sensory and motor, neuropathy, tumefactive, brain, demyelination Multifokal edinilmiş demiyelinizan duysal ve motor (MADSAM) nöropati, asimetrik motor ve duysal kayıplar ve periferik sinirlerde ileti blokları ile karakterizedir. Periferik demiyelinizan hastalıklara santral sinir sisteminde de demiyelinizasyonun eşlik edebildiği iyi bilinmektedir. Bu raporda, beyinde tümefaktif tek bir demiyelinizan lezyonun bulunduğu bir MADSAM olgusu sunulmaktadır. Yeni gelişen görme alanı defekti nedeniyle yapılan nörogörüntüleme sonucunda sağ parietooksipital bölgede başlangıçta tümör olarak yanlış tanı alan bir lezyon saptanmıştır. Patolojik inceleme sonucunda bu lezyonun demiyelinizan nitelikte olduğu anlaşılmıştır. Bu bulgudan iki yıl sonra, hastada periferik nöropatiyi düşündüren semptomlar ortaya çıkmış ve hasta MADSAM tanısı almıştır. Steroid tedavisi ile belirgin klinik ve elektrofizyolojik yanıt elde edilmiştir. Beyinde tümefaktif demiyelinizan lezyonlar, daha önce bir kronik enflamatuvar demiyelinizan nöropati olgusunda bildirilmiş olmasına karşın, MADSAM için bu tür bir bildirimde bulunulmamıştır. MADSAM olgularında da bu nedenle santral sinir sistemi tutulumu olasılığı gözden geçirilmelidir.

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“…Previous reports have pointed to an association between Hashimoto's thyroiditis and autoimmune demyelinating neuropathies such as Guillain–Barré syndrome, chronic inflammatory demyelinating polyneuropathy, and multifocal motor neuropathy, likely due to an underlying shared genetic susceptibility to autoimmune conditions [ 18 – 22 ]. The significant clinical improvement in our patient's distal weakness together with partial improvement in his NCS findings after 12 months of thyroxine therapy and the absence of other systemic involvements make the presence of an autoimmune demyelinating neuropathy a less likely diagnosis.…”

Section: Discussionmentioning

confidence: 99%

Severe Hypothyroidism Complicated by Myopathy and Neuropathy with Atypical Demyelinating Features

Brzozowska

Banthia

Thompson

et al. 2021

Case Reports in Endocrinology

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Autoimmune hypothyroidism may result in a wide range of neuromuscular disorders. The frequently observed neurological manifestations of acquired hypothyroidism include mild to moderate myopathy and sensorimotor neuropathy, which usually resolve by clinical and electrophysiological criteria, in adults treated with thyroid hormone replacement. We report a case of a 30-year-old male with severe hypothyroidism secondary to chronic autoimmune thyroiditis who presented with a 2-year history of progressive fatigue, upper and lower limb weakness, myalgia, and intermittent paraesthesia. His neurological exam demonstrated proximal and distal muscle weakness, lower limb areflexia, and relatively intact sensory modalities. The patient’s biochemistry revealed unusually and profoundly raised the thyroid stimulating hormone (TSH) level of 405.5 mIU/L (reference range (RR): 0.27–4.2 mIU/L) and creatine kinase (CK) level of 20,804 U/L (RR: 45–250 U/L), while his nerve conduction studies (NCS) demonstrated severe sensorimotor polyneuropathy with both axonal and demyelinating features. Thyroid hormone replacement therapy over the first 3 months resulted in biochemical normalization of his extremely deranged thyroid function tests (TFTs) and CK levels. At 12 months, despite maintaining euthyroidism and noticeable improvement in strength, his nerve conduction studies (NCS) demonstrated the continued absence of distal motor and sensory responses in his lower limbs with only partial improvement in sensory amplitudes and conduction velocities in his upper limbs. This report highlights the potential for severe neuromuscular consequences from advanced and chronic autoimmune hypothyroidism. The patient’s myopathy has resolved over a period of three months with prompt normalization of CK levels. Concerningly, the patient achieved significant but incomplete recovery from his mixed axonal and demyelinating neuropathy with residual mild distal weakness and areflexia in his lower limbs and persistent motor and sensory impairments on his NCS. The severity and incomplete resolution of our patient’s neurological manifestations emphasize the importance of early diagnosis and the need for prompt therapeutic intervention for hypothyroidism.

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Early neurophysiological evolution of chronic inflammatory demyelinating polyneuropathy in a patient with Hashimoto's thyroiditis

Cited by 13 publications

References 23 publications

Pediatric Hashimoto's encephalopathy with peripheral nervous system involvement

Pediatric Hashimoto's encephalopathy with peripheral nervous system involvement

Tumefactive Brain Demyelination Accompanying MADSAM Neuropathy

Severe Hypothyroidism Complicated by Myopathy and Neuropathy with Atypical Demyelinating Features

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