Postoperative neurological aggravation after anesthesia with sevoflurane in a patient with xeroderma pigmentosum: a case report

Journal of Medical Case Reports

Nordine Drissi Kamili¹  Hicham Azendour¹  Elhoucine Boutayeb¹  Najib Bouhabba¹  Bahija Yafat¹  Mustapha Bensghir¹  Salaheddine Fjouji¹ 

[1] Department of Anaesthesiology, Military Hospital Med V Rabat, University of Med V Souissi, Rabat, Morocco

关键词: Neurological worsening;    Sevoflurane;    General anesthesia;    Xeroderma pigmentosum;   

Others  :  1181246
DOI  :  10.1186/1752-1947-7-73

 received in 2012-05-13, accepted in 2013-02-14,  发布年份 2013

【 摘 要 】

Introduction

Xeroderma pigmentosum is a rare autosomal recessive disease that causes changes in skin pigmentation, precancerous lesions and neurological abnormalities. It is a defect in the nucleotide excision repair mechanism. It has been reported that volatile anesthetics has a possible genotoxic side effect and deranged nucleotide excision repair in cells obtained from a patient with xeroderma pigmentosum.

We report an unusual case of postoperative neurological aggravation in a patient with xeroderma pigmentosum anesthetized with sevoflurane.

Case presentation

A 24-year-old African woman, who has had xeroderma pigmentosum since childhood, was admitted to our hospital for a femoral neck fracture. A preoperative physical examination revealed that she had a resting tremor with ataxia. She had cutaneous lesions such as keratosis and hyperpigmentation on her face and both hands. There was no major alteration of cognitive function, muscular strength was maintained and her osteotendinous reflexes were preserved. Surgical fixation was performed under general anesthesia after the failure of spinal anesthesia. All parameters were stable during surgery. When she woke up four hours later, the patient presented with confusion and psychomotor agitation, sharpened reflexes and the Babinski reflex was present. Her postoperative test results and a magnetic resonance imaging scan were unremarkable. It was suggested that sevoflurane had had a probable deleterious effect on the neurological status of this patient.

Conclusion

The anesthetizing of a patient with xeroderma pigmentosum is associated with a risk of worsening neurological disorders. At present, there are no clear recommendations to avoid the use of volatile agents in the anesthetic management of patients with xeroderma pigmentosum. More clinical and experimental research is needed to confirm the sensitivity of patients with xeroderma pigmentosum to sevoflurane and other halogenated anesthetics.

【 授权许可】

   
2013 Fjouji et al.; licensee BioMed Central Ltd.

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