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Bilateral TKA for Charcot arthropathy associated with multiple levels of spinal canal stenoses: A case report and review of literature.

ABSTRACT: We report a case of an 84-year-old male with multiple levels of canal stenoses who are diagnosed as Charcot arthropathy of bilateral knees. Although he did not feel any pain, recurrence of joint fluid collection and severe instability prevented him from standing and walking. Deficit in superficial sensation and bathyesthesia below the level of Th10, and tendon reflexes of extremities were seen. Romberg test was positive. Laboratory data showed no abnormal findings. We performed bilateral TKA based on the diagnosis of Charcot arthropathy. At 3-year follow-up, there is no complaint about joint instability and collection of joint fluid.

SUBMITTER: Hayashi S

PROVIDER: S-EPMC5094381 | biostudies-literature | 2017 Mar

REPOSITORIES: biostudies-literature

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Bilateral TKA for Charcot arthropathy associated with multiple levels of spinal canal stenoses: A case report and review of literature.

Hayashi Seiju S Okada Tomonari T Horita Keiji K

Journal of orthopaedics 20161028 1

We report a case of an 84-year-old male with multiple levels of canal stenoses who are diagnosed as Charcot arthropathy of bilateral knees. Although he did not feel any pain, recurrence of joint fluid collection and severe instability prevented him from standing and walking. Deficit in superficial sensation and bathyesthesia below the level of Th10, and tendon reflexes of extremities were seen. Romberg test was positive. Laboratory data showed no abnormal findings. We performed bilateral TKA bas ...[more]

PMID: 27829729

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Project description:BackgroundClassic cerebrotendinous xanthomatosis (CTX; OMIM #213700) manifests with chronic diarrhea, juvenile cataracts, tendon xanthomas and neurological symptoms. It is due to biallelic inactivation of CYP27A1 wich leads to cholestanol accumulation in the central nervous system, eyes and tendons. Less commonly, the disease can present in young adults as spastic paraparesis in the absence of xanthomas.Case presentationWe report a 38-year old woman who presented with chronic diarrhea and progressive spastic paraparesis in her twenties. Brain magnetic resonance imaging (MRI) showed cerebral atrophy with diffuse periventricular white matter hyperintensities. Spinal MRI was normal. CYP27A1 gene sequencing confirmed the diagnosis of CTX. Chenodeoxycholic acid (CDCA) treatment was introduced with remission of diarrhea. Unfortunately, the treatment had to be discontinued several times and the patient developed psychosis and an severe ataxospastic gait. Spinal MRI revealed new linear hyperintensities of the corticospinal and gracile tracts. Thirty-three spinal CTX patients were identified by searching in Pubmed, EMBASE™ and Web of Science databases. All patients presented pyramidal signs and 48% had dorsal column signs. Juvenile cataracts were described in 78% of patients, chronic diarrhea in 65%, and tendon xanthomas in 31%. Disease improvement or stabilization with chenodeoxycholic acid was observed in 69% of patients. A higher prevalence of the Arg395Cys allele was observed in patients with spinal CTX as compared to CTX in general (ᵡ2; p < 0.00001).ConclusionsThe diagnosis of spinal CTX can be easily missed or delayed in absence of xanthomas. There is a higher prevalence of the Arg395Cys allele in spinal CTX as compared to classic childhood-onset CTX. CDCA treatment seems to stabilize or improve clinical symptoms in most patients. However, as seen in our patient and in two previously reported cases, sudden interruption of CDCA may lead to irreversible neurological complications.

Dataset Information

Bilateral TKA for Charcot arthropathy associated with multiple levels of spinal canal stenoses: A case report and review of literature.

Publications

Bilateral TKA for Charcot arthropathy associated with multiple levels of spinal canal stenoses: A case report and review of literature.

Similar Datasets

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