a) Studies with large populations
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Kolleri et al.1717. Kolleri JJ, Sasidharanpillai S, Vadakkayil B, Chathoth AT. A 10-year retrospective descriptive study on pure neuritic leprosy from a tertiary referral centre. Indian Dermatology Online J. 2019;10(1):13‐8. https://doi.org/10.4103/idoj.IDOJ_118_18 https://doi.org/10.4103/idoj.IDOJ_118_18...
, 2019, India |
A 10-year Retrospective Descriptive Study on Pure Neuritic Leprosy from a Tertiary Referral Centre. |
Retrospective descriptive study |
n=879 |
Delay in the diagnosis of pure neuritic leprosy due to the absence of dermatological lesions. Need for the training of health professionals for early detection of the disease. |
Medeiros et al.1919. Medeiros MF, Jardim MR, Vital RT, Nery JA, Sales AM, Moraes MO, et al. An attempt to improve pure neural leprosy diagnosis using immunohistochemistry tests in peripheral nerve biopsy specimens. Appl Immunohistochem Mol Morphol. 2014;22(3):222-30. https://doi.org/10.1097/PAI.0b013e31828dc70c https://doi.org/10.1097/PAI.0b013e31828d...
, 2014, Brazil |
An attempt to improve pure neural leprosy diagnosis using immunohistochemistry tests in peripheral nerve biopsy specimens. |
Experimental study |
n=28 Group cases (23) Control group (5) |
The immunohistochemical technique suggests the presence of immunoreactivity to specific anti-LAM and anti-PGL-1 antibodies as an additional tool in the diagnosis of pure neural leprosy. Immunohistochemical techniques can contribute to the early diagnosis of the disease. |
Bathala et al.2222. Bathala L, Krishnam VN, Kumar HK, Neladimmanahally V, Nagaraju U, Kumar HM, et al. Extensive sonographic ulnar nerve enlargement above the medial epicondyle is a characteristic sign in Hansen's neuropathy. PLoS Negl Trop Dis. 2017;11(7):e0005766. https://doi.org/10.1371/journal.pntd.0005766 https://doi.org/10.1371/journal.pntd.000...
, 2017, India |
Extensive sonographic ulnar nerve enlargement above the medial epicondyle is a characteristic sign in Hansen's neuropathy. |
Experimental study |
n=48 Group cases (18) Control group (30) |
Patients with ulnar neuropathy had a unique pattern of nerve enlargement on ultrasound examination. It starts at the ulnar sulcus and can increase up to four centimeters above the medial epicondyle. |
Jaiswal et al.2323. Jaiswal N, Chakraborti S, Nayak K, Pai S, Shelley BP, Sreeram S, et al. Hansen's neuritis revisited: a clinicopathological study. J Neurosci Rural Pract. 2018;9(1):42‐55. https://doi.org/10.4103/jnrp.jnrp_438_17 https://doi.org/10.4103/jnrp.jnrp_438_17...
, 2018, India |
Hansen's Neuritis Revisited – A Clinicopathological Study. |
Retrospective study |
n=81,013 (histopathological examination samples) |
The common clinical presentation of Hansen's neuritis is mononeuritis multiplex. Neurological examination and nerve biopsy are recommended for diagnosis. |
Antunes et al.2424. Antunes SL, Chimelli L, Jardim MR, Vital RT, Nery JA, Corte-Real S, et al. Histopathological examination of nerve samples from pure neural leprosy patients: obtaining maximum information to improve diagnostic efficiency. Mem Inst Oswaldo Cruz. 2012;107(2):570. https://doi.org/10.1590/s0074-02762012000200015 https://doi.org/10.1590/s0074-0276201200...
, 2012, Brazil |
Histopathological examination of nerve samples from pure neural leprosy patients: obtaining maximum information to improve diagnostic efficiency. |
Experimental study |
n=340 (144 with pure neuritic leprosy and 196 with non-leprosy peripheral neuropathies) |
The study demonstrates the importance of performing nerve biopsy exams for the identification of leprosy neuropathy, with a predominance of non-inflammatory histopathological changes in pure neuritic leprosy samples. |
Kulshreshtha et al.2828. Kulshreshtha D, Malhotra KP, Malhotra HS, Thacker AK, Garg RK, Singh AK, et al. Mandating nerve biopsy: a step towards personalizing therapy in pure neuritic leprosy. J Peripher Nerv Syst. 2018;23(3):190‐6. https://doi.org/10.1111/jns.12283 https://doi.org/10.1111/jns.12283...
, 2018, India |
Mandating nerve biopsy: A step towards personalizing therapy in pure neuritic leprosy. |
Retrospective study |
n=78 |
Predominance of the ulnar nerve. Only 16 of the 38 patients demonstrated bacilli in Wade-Fite staining. |
Shukla et al.3232. Shukla B, Verma R, Kumar V, Kumar M, Malhotra KP, Garg RK, et al. Pathological, ultrasonographic, and electrophysiological characterization of clinically diagnosed cases of pure neuritic leprosy. J Peripher Nerv Syst. 2020;25(2):191-203. https://doi.org/10.1111/jns.12372 https://doi.org/10.1111/jns.12372...
, 2020, India |
Pathological, ultrasonographic, and electrophysiological characterization of clinically diagnosed cases of pure neuritic leprosy. |
Observational study |
n=100 |
Ultrasound, histological, and electrophysiological evaluation, taken together, constitute the best method for diagnosis of pure neural leprosy, showing greater accuracy and ability to exclude or confirm differential diagnoses. |
De et al.3838. De A, Reja AHH, 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(6):635‐43. https://doi.org/10.4103/ijd.IJD_115_17 https://doi.org/10.4103/ijd.IJD_115_17...
, 2017, India |
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. |
Observational study |
n=62 |
The use of fine-needle aspiration cytology in combination with Ziehl-Neelsen staining and PCR is a quick, simple and easy solution that can be used for the diagnosis of pure neural leprosy. |
Rodriguez et al.3535. Rodriguez G, Pinto R, Gomez Y, Rengifo ML, Estrada OL, Sarmiento M, et al. Pure neuritic leprosy in patients from a high endemic region of Colombia. Lepr Rev. 2013;84(1):41‐50. PMID: 23741881, 2013, Colombia |
Pure neuritic leprosy in patients from a high endemic region of Colombia. |
Descriptive observational study |
n=36 |
21 of the 36 individuals initially suspected of pure neural leprosy were confirmed, based mainly on epidemiological and clinical findings, and on changes in amplitude and nerve conduction on electroneuromyography. Five of them had positive PCR for M. leprae DNA on nerve biopsy. |
b) Case report studies
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Freitas et al.1111. Freitas RCS, Pereira CAZ, Muri NT, Franzon VAZ, Fatuch CAC. Primary neural leprosy mimicking rheumatological disorders. Rev Assoc Med Bras. 2019;65(6):767-70. https://doi.org/10.1590/0004-282X20130046 https://doi.org/10.1590/0004-282X2013004...
, 2019, Brazil |
Primary Neural Leprosy mimicking rheumatological disorders |
Case report |
n=1 |
Clinicians who are alert and trained to recognize this clinical form of leprosy are necessary. Correct referral to reference centers accelerates the investigation, contributing towards early diagnosis, correct classification, and treatment, preventing irreversible sequelae with severe functional disability. |
Serrano-Coll et al.1818. Serrano-Coll H, Mieles O, Escorcia C, Díaz A, Beltrán C, Cardona-Castro N. A case series of pure neural leprosy in patients diagnosed in a specialized center for the control of Hansen's disease in Colombia. Biomedica. 2018;38(2):153‐61. https://doi.org/10.7705/biomedica.v38i0.3690 https://doi.org/10.7705/biomedica.v38i0....
, 2018, Colombia |
A case series of pure neural leprosy in patients diagnosed in a specialized center for the control of Hansen's disease in Colombia. |
Case report |
n=4 |
There is a need for advancement of the development of a diagnostic method, using serological and genomic mechanisms. Health professionals should mandatorily include peripheral neuropathy as a differential diagnosis of leprosy disease. |
Santos et al.2020. Santos DFD, Pereira RC, Goulart IMB. Anterior tarsal tunnel syndrome: an atypical involvement in primary neural leprosy. Arq. Neuropsiquiatr. 2019;77(10):754-5. https://doi.org/10.1590/0004-282X20190114 https://doi.org/10.1590/0004-282X2019011...
, 2019, Brazil |
Anterior tarsal tunnel syndrome: an atypical involvement in primary neural leprosy. |
Case report |
n=1 |
The anterior tarsal syndrome can be a finding of primary neural leprosy, even if it is atypical. It is necessary to include leprosy as one of the differential diagnoses of anterior tarsus syndrome. |
Omar & Hussein2121. Omar AE, Hussein MR. Clinically unsuspected neuritic leprosy with caseation necrosis. Ultrastruct Pathol. 2012;36(6):377‐80. https://doi.org/10.3109/01913123.2012.700390 https://doi.org/10.3109/01913123.2012.70...
, 2012, Egypt |
Clinically unsuspected neuritic leprosy with caseation necrosis. |
Case report |
n=1 |
Caseation necrosis (abscess) can be a clinical finding, although atypical and rare, of neuritic leprosy. Mass lesions with necrosis, when found in biopsy samples, must be taken into account for discussing a possible case of leprosy. |
Jaramillo et al., 20162525. Jaramillo L, Giraldo N, Arboleda M, Rodríguez G. Lepra neural pura de 18 años de evolución. Infect. 2017;21(1):55-60. https://doi.org/10.22354/in.v21i1.642 https://doi.org/10.22354/in.v21i1.642...
Colombia |
Lepra neural pura de 18 años de evolución. |
Case report |
n=1 |
Pure neural leprosy remains a challenge for healthcare professionals. |
Pardal-Fernández et al.2626. Pardal-Fernandez JM, Ezsol-Lendvai S, Rodríguez-Vazquez M, Agudo-Mena JL, Godes-Medrano B. Pure neural leprosy. Diagnostic aspects of a clinical case. Rev Neurol. 2016;63(6):257-61. PMID: 27600740, 2016, Spain |
Lepra neural pura. Aspectos diagnosticos en un caso clinico |
Case report |
n=1 |
Early diagnosis and treatment of the disease allowed for better patient recovery. |
Beltrame et al.2727. Beltrame A, Barabino G, Cicciò C, Monteiro GB, Cavalchini A, Carbognin G, et al. Magnetic resonance imaging in pure neural leprosy. Int J Infect Dis. 2017;60:42‐3. https://doi.org/10.1016/j.ijid.2017.04.022 https://doi.org/10.1016/j.ijid.2017.04.0...
, 2017, Italy |
Magnetic resonance imaging in pure neural leprosy |
Case report |
n=1 |
Health professionals have few techniques to analyze the signs of leprosy. Magnetic resonance imaging can be an auxiliary tool in diagnosis. |
Rai et al.2929. Rai D, Malhotra HS, Garg RK, Goel MM, Malhotra KP, Kumar V, et al. Nerve abscess in primary neuritic leprosy. Lepr Rev. 2013;84(2):136‐40. PMID: 24171239, 2013, India |
Nerve abscess in primary neuritic leprosy. |
Case report |
n=1 |
Caseous necrosis of nerve injuries occasionally coalesces to form a nervous abscess. Data suggest that abscess is more common in borderline tuberculoid leprosy. Ultrasound seems to be the best diagnostic technique. |
Gupta et al.3030. Gupta V, Dev T, Das CJ, Khanna N. Nerve abscess in pure neural leprosy mistaken for peripheral nerve sheath tumor with disastrous consequence: what can we learn? BMJ Case Rep. 2017;2017:bcr2017221023. https://doi.org/10.1136/bcr-2017-221023 https://doi.org/10.1136/bcr-2017-221023...
, 2017, India |
Nerve abscess in pure neural leprosy mistaken for peripheral nerve sheath tumor with disastrous consequence: what can we learn? |
Case report |
n=1 |
Diagnosing pure neural leprosy is a challenge for doctors. Presence of nerve abscess without thickening of peripheral nerves. Among the non-invasive methods, ultrasound seems to be the best method for diagnosis. |
Shrestha et al.3131. Shrestha BK, Ranabhat K, Pant R, Sapkota S, Shrestha S. Neuritic leprosy: an intriguing re-visit to a forbidden ailment. Kathmandu Univ Med J (KUMJ). 2019;17(65):73‐6. PMID: 31734684, 2019, Nepal |
Neuritic Leprosy; An Intriguing Re-visit to a Forbidden Ailment. |
Case report |
n=1 |
Pure neuritic leprosy, although rare, should be considered as a differential diagnosis in cases where there is a presentation of peripheral neuropathy in endemic areas of leprosy. Prompt diagnosis and treatment are imperative to avoid permanent neurological damage. |
Pradhan et al.3333. Pradhan S, Padhi T, Panda BB, Nayak BP. Polyneuritic variant of pure neuritic leprosy with extensive involvement of peripheral nerves and sparing of the polio-affected limb: a rare case report. J Dtsch Dermatol Ges. 2016;14(7):730‐3. https://doi.org/10.1111/ddg.12697 https://doi.org/10.1111/ddg.12697...
, 2016, India |
Polyneuritic variant of pure neuritic leprosy with extensive involvement of peripheral nerves and sparing of the polio-affected limb: a rare case report. |
Case report |
n=1 |
The association of poliomyelitis and leprosy in the same patient showed that a “protection” of the nerves affected by poliomyelitis, which were not affected by leprosy may be possible. |
Karjigi et al.3434. Karjigi S, Herakal K, Murthy SC, Bathina A, Kusuma MR, Nikhil KR. Primary neuritic Hansen's disease presenting as ulnar nerve abscess in a human immunodeficiency virus-positive patient. Indian J Lepr. 2015;87(3):169‐74. PMID: 26999990, 2015, India |
Primary Neuritic Hansen's Disease presenting as Ulnar Nerve Abscess in a Human Immunodeficiency Virus Positive Patient. |
Case report |
n=1 |
Coinfection between HIV and leprosy can result in atypical manifestations. The patient also had a silent ulnar nerve abscess, diagnosed by ultrasound. |
Payne et al.3636. Payne R, Baccon J, Dossett J, Scollard D, Byler D, Patel A, et al. Pure neuritic leprosy presenting as ulnar nerve neuropathy: a case report of electrodiagnostic, radiographic, and histopathological findings. J Neurosurg. 2015;123(5):1238‐43. https://doi.org/10.3171/2014.9.JNS142210 https://doi.org/10.3171/2014.9.JNS142210...
, 2015, United States of America |
Pure neuritic leprosy presenting as ulnar nerve neuropathy: a case report of electrodiagnostic, radiographic, and histopathological findings. |
Case report |
n=1 |
Pure neural leprosy is a challenging diagnosis. Delayed diagnosis can lead to permanent neural damage. |
Almeida Neto et al.3737. Almeida Neto FB, Aureliano Neto R, Cardoso LMC. The first report of pure neuritic leprosy with involvement of the anterior femoral cutaneous nerve. Neurol Int. 2019;11(2):8001. https://doi.org/10.4081/ni.2019.8001 https://doi.org/10.4081/ni.2019.8001...
, 2019, Brazil |
The first report of pure neuritic leprosy with involvement of the anterior femoral cutaneous nerve. |
Case report |
n=1 |
This is the first case report involving the superficial femoral cutaneous nerve as a manifestation of leprosy. Pure neural leprosy is a diagnostic challenge. In endemic countries, any clinical manifestation involving the peripheral nervous system should be considered as a possible case of leprosy. |