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E-IJD SHORT COMMUNICATION
Year : 2015  |  Volume : 60  |  Issue : 2  |  Page : 213
Isolated tenosynovitis as a sole manifestation: The great mimicker still continues to surprise Us


1 Department of Dermatology, Institute of Post Graduate Medical Education and Research (IPGMER), Kolkata, West Bengal, India
2 Department of Biochemistry, Institute of Post Graduate Medical Education and Research (IPGMER), Kolkata, West Bengal, India

Date of Web Publication3-Mar-2015

Correspondence Address:
Abhishek De
Department of Dermatology, IPGMER, 242, AJC Bose Road, Bhowanipur, Kolkata - 700020, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.152577

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   Abstract 

A middle aged male presented with non-tender cystic swelling over left distal forearm since 1 year. No other cutaneous abnormality could be found except mild paresthesia of the overlying skin and equivocal thickening of the ipsilateral ulnar nerve. Routine investigation was within normal limits. Detailed workup of the patient including MRI of the lesion suggested the diagnosis as tenosynovitis with a soft tissue mass. Fine needle aspiration cytology from the cyst showed foamy macrophages and acid fast bacilli; while PCR of the aspirate confirmed the etiological agent as M. leprae. We, thus, report a unique case of isolated tenosynovitis as a sole manifestation of pure neural leprosy which is extremely rare in world literature.


Keywords: PCR, pure neuritic leprosy, tenosynovitis


How to cite this article:
De A, Surana TV, Biswas S, Reja AH, Chatterjee G. Isolated tenosynovitis as a sole manifestation: The great mimicker still continues to surprise Us. Indian J Dermatol 2015;60:213

How to cite this URL:
De A, Surana TV, Biswas S, Reja AH, Chatterjee G. Isolated tenosynovitis as a sole manifestation: The great mimicker still continues to surprise Us. Indian J Dermatol [serial online] 2015 [cited 2020 Apr 1];60:213. Available from: http://www.e-ijd.org/text.asp?2015/60/2/213/152577

What was known?

  1. Rheumatological manifestation including tenosynovitis can be a part of the clinical spectrum of leprosy.
  2. Isolated tenosynovitis has only once been reported in literature; however, etiology of that case was not confirmed with any specific investigation.



   Introduction Top


Leprosy is a chronic granulomatous infectious disease caused by Mycobacterium leprae with predominant involvement of skin and peripheral nervous system. [1] Involvement of musculoskeletal system is the third most common manifestation. Articular involvement in leprosy has been reported in ancient Chinese literature. [2] Acute onset painful edema of hands with arthritis and tenosynovitis with or without lepra reaction are included in the spectrum of rheumatologic manifestations of leprosy. [3],[4]


   Case Report Top


A middle aged male presented with non-tender cystic swelling over left distal forearm since 1 year which was insidious in onset and gradually progressing [Figure 1]. At presentation the lesion was 2 × 2 cm, fixed to overlying skin but mobile in transverse direction over underlying structure. On detailed examination, no other cutaneous abnormality could be found barring mild paresthesia of the overlying skin and equivocal thickening of the ipsilateral ulnar nerve. Routine investigation revealed no abnormality. X-ray wrist joint was within normal limits.
Figure 1: Subcutaneous swelling over volar aspect of the left wrist joint

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Fine needle aspiration from the cystic swelling obtained purulent material which on cytology after H and E stain showed plenty of neutrophils, few epitheloid cells and few foamy macrophages [Figure 2]. Modified Z-N stain demonstrated acid fast bacilli [Figure 3]. MRI scan of the lesion revealed soft tissue swelling and confirmed the diagnosis of tenosynovitis. But for us, the dilemma remained regarding the etiology of tenosynovitis whether it is from Mycobacterium tuberculosis or M. leprae.
Figure 2: Photomicrograph of FNAC showing foamy macrophage in the dermis (H and E × 400)

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Figure 3: Modified Z-N stain of the aspirate from the lesion shows numerous acid fast bacilli

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We conducted nerve conduction study which revealed axonal neuropathy of the ipsilateral ulnar nerve. But still the diagnosis of pure neural leprosy (PNL) was doubtful. So, we decided to repeat the aspiration. PCR from the aspirate detected M. leprae specific DNA with a single band at 194bp which confirmed the etiology as M. leprae [Figure 4].
Figure 4: PCR from the aspirate detected M. leprae specific DNA with a single band at 194bp

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   Discussion Top


In endemic areas, predominant or sole rheumatic presentation can make the diagnosis of leprosy difficult in the absence of typical cutaneous manifestations. Leprosy can present as arthritis, tenosynovitis and enthesitis in varied combination. Acute arthritis in leprosy usually occurs as a part of lepra reaction or by direct infiltration in the synovium by lepra bacilli. [1]

Tenosynovitis in leprosy has been the subject of anecdotal reports. [2],[3] A combination of arthritis, tenosynovitis with or without paresthesia and thickened nerve is highly suggestive of leprosy. Isolated tenosynovitis has only once been reported in literature; however, etiology of that case was not confirmed with any specific investigation. [2]

Pure neuritic cases are characterized by neuropathic symptoms and thickened nerves with or without motor or sensory loss, in the absence of any cutaneous sign of leprosy. [4] Arthritis in pure neuritic cases was reported previously [5] but no detailed clinical or laboratory data was provided. Isolated tenosynovitis without arthritis in a case of pure neuritic leprosy is rare in world literature.

Radiological abnormalities in patients of arthritis due to leprosy can range from normal joint to subluxation and complete destruction. Juxta-articular erosions quite similar to rheumatoid arthritis was also reported. [5],[6] Lack of these features in our case helped us to exclude arthritis.

PCR for detection of M. leprae DNA-encoding-specific genes or repeat sequences is potentially highly sensitive and specific method for detection of M. leprae DNA in both multi-bacillary and paucibacillary cases. [8] We could only come to a specific diagnosis in this unusual case by confirming the diagnosis with PCR as demonstration of acid fast bacilli alone is not sufficient to rule out tuberculosis as etiological agent of tenosynovitis.

This makes our case unique as the dilemma remained in the diagnosis until we did PCR which confirmed the etiology as M. leprae.

Thus, even in the era of sophisticated diagnostic aids and tall claims of leprosy being eradicated, this great mimicker still continues to surprise us. This is the first case of isolated tenosynovitis presenting as a sole manifestation of Pure Neural Hansen which was confirmed by PCR. [9] We believe that patients presenting with an isolated tenosynovitis can present a serious diagnostic challenge and a highly vigilant mind along with sophisticated diagnostic aides can reveal the final diagnosis.


   Conclusion Top


Tenosynovitis is one of the rare manifestations in pure neuritic leprosy usually reported along with arthritis. Isolated tenosynovitis had only been reported once in literature.This is first case of isolated tenosynovitis as a sole manifestation of pure neuritic leprosy in which the etiological agent is confirmed with specific diagnostic investigations including PCR.

 
   References Top

1.
Britton WJ, Lockwood DN. Leprosy. Lancet 2004;363:1209-19.  Back to cited text no. 1
    
2.
Messner RP. Arthritis due to mycobacteria, fungi and parasites. In: Koopman WJ, McCarty DJ, editors. Arthritis and Allied Conditions. 13 th ed. Maryland: Williams and Wilkins; 1997. p. 2305-20.  Back to cited text no. 2
    
3.
Albert DA, Weisman MH, Kaplan R. The rheumatic manifestations of leprosy (Hansen disease). Medicine (Baltimore) 1980;59:442-8.  Back to cited text no. 3
    
4.
Haroon N, Agarwal V, Aggarwal A, Kumari N, Krishnani N, Misra R. Arthritis as presenting manifestation of pure neuritic leprosy: A rheumatologist's dilemma. Rheumatology (Oxford) 2007;46:653-6.  Back to cited text no. 4
    
5.
Atkin SL, el-Ghobarey A, Kamel M, Owen JP, Dick WC. Clinical and laboratory studies of arthritis in leprosy. Br Med J 1989;298:1423-5.  Back to cited text no. 5
    
6.
Noordeen SK. Epidemiology of (poly) neuritic type of leprosy. Lepr India 1972;44:90-6.  Back to cited text no. 6
    
7.
Modi TH, Lele RD. Acute joint manifestations in leprosy. J Assoc Physicians India 1969;17:247-54.  Back to cited text no. 7
    
8.
Williams DL, Gillis TP, Booth RJ, Looker D, Watson JD. The use of a specific DNA probe and polymerase chain reaction for the detection of Mycobacterium leprae. J Infect Dis 1990;162:193-200.  Back to cited text no. 8
    
9.
Agarwal V, Wakhlu A, Aggarwal A, Misra R. Tenosynovitis as the presenting manifestation of leprosy. J Indian Rheumatol Assoc 2002;10:69-70.  Back to cited text no. 9
    

What is new?

  1. Isolated tenosynovitis can be a presenting feature of Hansen disease.
  2. To the best of our knowledge this is first case of isolated tenosynovitis as a sole manifestation of pure neuritic leprosy in which the etiological agent is confirmed with specific diagnostic investigations including PCR.


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    Abstract
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   Discussion
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    References
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