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A new diagnostic sampling method in pure neural leprosy: the scraping of the myelin sheath☆☆ ☆☆ Study conducted at the Ospedale Policlinico San Martino, Largo Rosanna Benzi, Genoa, Italy.

Dear Editor,

Pure Neural Leprosy (PNL) is a form of leprosy characterized by neural involvement without any skin lesions.11 de Freitas MR, Said G. Leprous neuropathy. Handb Clin Neurol. 2013;115:499-514. PNL affects about 3%–10% of patients with leprosy and it can occur in any spectrum, although it is more frequent in the tuberculoid type.22 Girdhar BK. Neuritic leprosy. Indian J Lepr. 1996;68:35-42. We present a case of a patient affected by PNL diagnosed through the scraping of myelin sheaths of the ulnar nerve, Ziehl-Neelsen (ZN) staining, and Polymerase Chain Reaction (PCR).

A 78-year-old man, a professional missioner in the Philippines and Papua New Guinea, has presented sensory loss (touch, pain, and temperature) of left foot and pain of left hand present over a period of 4 years. A physical examination revealed dorsal flexion deficit of the left foot, superficial paraesthesia, and dysesthesia of toes associated with impaired deep sensitivity. In addition, he presented paraesthesia and dysesthesia to the IV and V fingers of the left hand. The left ulnar nerve was palpable and enlarged on the left elbow and no cutaneous lesions were found. The research of Acid-Fast Bacillus (AFBs) in the nasal swab and the slit skin smears from earlobes and left elbow was negative. Motor and sensory action potential of the left ulnar nerve, left peroneal nerve, left anterior and posterior tibial nerves are suggestive of mono-neuritis multiplex. Magnetic Resonance Imaging (MRI) of the left elbow showed the enlarged ulnar nerve partially damaged by entrapment within the fibro-osseous tunnel. Neurosurgery allowed the debridement of the ulnar nerve and, at the same time, the scraping of the perineural tissue. ZN stain and PCR of the scraping were positive for the presence of M. leprae and the diagnosis of tuberculoid leprae with PNL was made. Antibodies against phenolic glycolipid-1 antigen (anti-PGL antibody) were negative. A therapy based on a combination of three drugs (rifampicin 600 mg once a month, dapsone 100 mg daily, and clofazimine 300 mg once a month and 50 mg daily) associated with prednisone 25 mg and gabapentin 300 mg (2 cp/die) was started with improvement of symptoms.

To the best of our knowledge, this is the first case of PNL diagnosed through scraping, ZN staining, and PCR test. Scraping is a technique that allows obtaining a clinical specimen rubbing a part of the body, in our case myelin sheaths of a nerve. The surface is scraped with a 15 Bard-Paker blade held at a right angle to the incision. Upon scraping, perineural tissue is obtained and examined by ZN staining and PCR test. Traditionally, diagnostic criteria for the diagnosis of PNL consist of nerve tissue samples obtained out of a nerve biopsy, analysis of PCR, and measure of anti-PGL-1 antibody levels.33 Jardim MR, Antunes SL, Santos AR, Nascimento OJ, Nery JA, Sales AM, et al. Criteria for diagnosis of pure neural leprosy. J Neurol. 2003;250:806-9. However, the invasive procedure of nerve biopsy was criticized by Abhishek De et al. because it has a high rate of complications.44 Reja AH, De A, Biswas S, Chattopadhyay A, Chatterjee G, Bhattacharya B, et al. Use of fine needle aspirate from peripheral nerves of pure-neural leprosy for cytology and PCR to confirm the diagnosis: a pilot study. Indian J Dermatol Venereol Leprol. 2013;79:789-94. They proposed a simple technique of FNAC coupled with PCR in a pilot study44 Reja AH, De A, Biswas S, Chattopadhyay A, Chatterjee G, Bhattacharya B, et al. Use of fine needle aspirate from peripheral nerves of pure-neural leprosy for cytology and PCR to confirm the diagnosis: a pilot study. Indian J Dermatol Venereol Leprol. 2013;79:789-94. and they confirmed its efficacy in a 4-year study.55 De A, Hasanoor Reja AH, Aggarwal I, Sen S, Sil A, Bhattacharya B, et al. Use of Fine Needle Aspirate from Peripheral Nerves of Pure-neural Leprosy for Cytology and Polymerase Chain Reaction to Confirm the Diagnosis: A Follow-up Study of 4 Years. Indian J Dermatol. 2017;62:635-43. In our case, we did not use the technique of FNAC because an invasive procedure, surgery, was required to solve the compression of the ulnar nerve in the cubital tunnel. However, the scraping of the myelin sheath is a simple tissue sampling method during surgical procedures with less risk of nerve damage.

  • ☆☆
    Study conducted at the Ospedale Policlinico San Martino, Largo Rosanna Benzi, Genoa, Italy.
  • Financial support
    None declared.

References

  • 1
    de Freitas MR, Said G. Leprous neuropathy. Handb Clin Neurol. 2013;115:499-514.
  • 2
    Girdhar BK. Neuritic leprosy. Indian J Lepr. 1996;68:35-42.
  • 3
    Jardim MR, Antunes SL, Santos AR, Nascimento OJ, Nery JA, Sales AM, et al. Criteria for diagnosis of pure neural leprosy. J Neurol. 2003;250:806-9.
  • 4
    Reja AH, De A, Biswas S, Chattopadhyay A, Chatterjee G, Bhattacharya B, et al. Use of fine needle aspirate from peripheral nerves of pure-neural leprosy for cytology and PCR to confirm the diagnosis: a pilot study. Indian J Dermatol Venereol Leprol. 2013;79:789-94.
  • 5
    De A, Hasanoor Reja AH, Aggarwal I, Sen S, Sil A, Bhattacharya B, et al. Use of Fine Needle Aspirate from Peripheral Nerves of Pure-neural Leprosy for Cytology and Polymerase Chain Reaction to Confirm the Diagnosis: A Follow-up Study of 4 Years. Indian J Dermatol. 2017;62:635-43.

Publication Dates

  • Publication in this collection
    26 July 2021
  • Date of issue
    May-Jun 2021

History

  • Received
    24 Apr 2020
  • Accepted
    31 May 2020
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