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Table of Contents   
CASE REPORT  
Year : 2016  |  Volume : 9  |  Issue : 6  |  Page : 396-399
Sporotrichosis of the upper limb in a post-traumatic patient: A case report


Department of Orthopedic, School of Medicine, Universiti Sains Malaysia, Kubang Kerian, Kelantan Malaysia

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Date of Web Publication14-Nov-2016
 

   Abstract 

Sporotricosis is a form of chronic lymphocutaneous infection that follows traumatic inoculation of the fungus Sporothrix schenckii into the skin leading to subcutaneous mycoses. History of atypical ulcer with cellulitis that did not respond to courses of antibiotics should draw the clinician's attention to other differential diagnosis such as sporotrichosis because it can mimic other cutaneous presentations as well such as cellulitis.

Keywords: Cellulitis, post-trauma, sporotrichosis

How to cite this article:
Ab-Rahman S, Ahmed-Shokria A. Sporotrichosis of the upper limb in a post-traumatic patient: A case report. Ann Trop Med Public Health 2016;9:396-9

How to cite this URL:
Ab-Rahman S, Ahmed-Shokria A. Sporotrichosis of the upper limb in a post-traumatic patient: A case report. Ann Trop Med Public Health [serial online] 2016 [cited 2017 Sep 20];9:396-9. Available from: http://www.atmph.org/text.asp?2016/9/6/396/193935

   Introduction Top


In 1986, Benjamin Schuck was the first to diagnose sporotrichosis in a patient with refractory chronic subcutaneous abscess.[1] Since then, many sporadic and endemic cases had been reported worldwide.

Sporotrichosis is one of the commonest subcutaneous mycoses; however, its presentation can mimic other cutaneous diseases such as cellulitis and can be missed even by trained professionals. Although, sporotrichosis is more common in the tropical or subtropical region, it is not exclusive only to these regions because cases have been reported in Japan and Eastern Europe as well.[1],[2],[3] The fungi can be found almost anywhere especially in soil, fresh water, and even in marine aquatic. Regular contact with pets such as cats or dogs is associated with this infection as well.

We report a case of a patient who was initially thought to have posttrauma ulcer with cellulitis of the right hand. The problem has not been fully resolved despite full courses of antibiotics from two different doctors.


   Case Report Top


A 30-year-old gentleman was referred to us by a general practitioner (GP) for a nonresolving cellulitis of the right hand and forearm for a 6-week duration. The patient gave a history of a motorbike accident before the onset of his problem. The patient did not sustain any other injury apart from a minor superficial wound over the dorsum of his right hand. The patient did not seek any medical treatment as he thought that his injury was nontrivial. After 3 days, he developed pain and redness over the dorsum of his right hand. In addition, he claimed that there was an on-off serous discharge from his wound associated with the swelling. After the injury, he denied contact with soils, untreated water source, or any other dirty environment. He does not have pets in his house.

Despite reasonable wound care by his wife, who works as an assistant nurse at a local clinic, the redness started to spread to his whole hand after 4 days. In addition, he claimed that he experienced a low-grade fever when the swelling started to ascend to his forearm. It was at this point, that he went to see a GP who diagnosed him for having cellulitis. The GP made him start a 1-week course of oral antibiotic.

Initially, the swelling, redness, and pain reduced slightly, but after a few days of stopping antibiotic, his condition worsened. After the fourth week of the onset, the patient saw another GP who diagnosed him for having cellulitis with lymphadenitis. Again, the patient was started with oral antibiotic for 1 week. The symptoms partially improved but a few days after stopping the antibiotic, the symptoms worsened again. This time, the patient saw the same GP who then referred this case to our center for further management.

Examination of the patient's right hand revealed a 1 cm × 3 cm healing wound over the dorsal region. There was no active discharge noted. His right hand, as well as his forearm was erythematous almost up to the elbow level. The erythema over the forearm followed a distinctive pattern. It spread along the lymphatic channels of the forearm. There was no other skin lesion or ulcer noted. There was tenderness over the erythematous area associated with multiple discrete swellings with sizes ranging from 2 mm to 5 mm. The nodules were tender, but not fluctuant [Figure 1]. The patient was able to move all his fingers and there was no restriction of movement over his wrist and elbow joints.
Figure 1: Healing primary lesion over the dorsum of the right hand with extensive cellulitis noted with a distinctive pattern of cutaneous lesion along the distribution of lymphatic channels of the right hand and the forearm

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A clinical diagnosis of nodular lymphangitis was made, based on history and clinical appearance of erythematous nodular swellings along the lymphatic channels. Fine needle aspiration was done to collect samples for histopathological examination (HPE) and bacteria culture. The sample was additionally sent for mycobacteria and fungal culture. The patient was then put on an empirical antifungal drug: oral itraconazole 200 mg three times daily [ter die sumendum (tds)]. There was marked improvement of his condition within 6 days of administration. In view of good clinical progress, the patient was discharged home after day 6 of admission with the instruction to continue with oral itraconazole 200 mg tds.

The HPE and culture results were obtained after the patient was discharged. The mycobacterium and bacteria culture was negative. The HPE was inconclusive. However, the fungal culture showed growth after day 6 of inoculation. The growth on Sabouraud agar showed filamentous mold that subsequently darkened as it matured. Microscopic examination revealed the dimorphic fungi Sprothrix schenckiii [Figure 2]a and [Figure 2]b. This result justifies our empirical antifungal management.
Figure 2: (a) Mature blackish fungal colony growth noted on the Sabouraud agar media (b) Microscopic examination of the mold form of Sporothrix schenckii (40× magnification)

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The patient was seen again at the outpatient clinic 4 weeks after discharge from the ward. The culture result was informed and discussed with the patient. On examination, most of his redness and swelling had disappeared and there was no more pain [Figure 3]a and [Figure 3]b. The patient was continued for another 2-week course of itraconazole.
Figure 3: (a and b) 4 weeks after commencing itraconazole. Most of the redness over the right hand and forearm had disappeared

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Itraconazole was continued for another 2 weeks to complete a total course of 6 weeks.

The patient was seen again at 3 months after ward discharge, at which he was completely clear of any signs or symptoms of nodular lymphangitis. The patient was discontinued from further clinical follow-up.


   Discussion Top


Nodular lymphangitis is a syndrome characterized by the development of superficial cutaneous lesions that progress along dermal and subcutaneous lymphatics. The disease usually presents with a strong history of direct cutaneous inoculation or trauma; such as thorn prick, wood splint, or other contaminated materials. The fungus Sporothrix schenckii is the main causative agent for nodular lymphangitis, hence the name sporotrichosis. Sporotrichosis is divided into four clinical categories:

  1. Lymphocutaneous,
  2. Fixed cutaneous,
  3. Multifocal or dessiminated, and
  4. Extracutaneous.


The lymphocutaneous variant of sporotrichosis that usually leads to the nodular lymphangitis, contributes to 75% of cases.[1],[3],[4] Although common, its presentation especially with concomitant swelling and erythema is often misdiagnosed and commonly confused with cellulitis, lymphangitis, and thrombophlebitis, which are typically caused by staphylococci andstreptococci.[3]

Sporothrix schenckii is found as mold in decaying vegetation, moss, soil, and other physical environments, where it has adapted, throughout the world. When this microorganism gains entry into host cells, they transform into budding yeast cells. The cells are capable of eliciting granulomatous infection, necrosis, and abscess formation.[5] The mode of entry besides subcutaneous inoculation or direct trauma, can as well be indirectly due to animal bites or scratches from domestic or wild animals, and even dolphins.[3] In addition, this fungus is common in aquatic environment such as sea water, fresh water such as rivers, and even aquariums. There is no specific age or sex distribution with even children being exposed to the disease.[3],[6],[7]

Clinically, patients usually present with initial painless erythematous papules. They develop 7 to 30 days after inoculation. Soon, there will be formation of ulcers and even ulceration. It is quite common to have serous or purulent discharge. The classical feature is the course of lesion following the lymphatic drainage ascending proximally. Often, there are palpable regional lymph nodes. The extracutaneous form can manifest as osteoarticular infection, monoarthritis, tenosynovitis, pulmonary sporotrichosis, sporotrichosis meningitis, and ocular sporotrichosis. Systemic symptoms are very rare except in immunocompromised patients [2] Although, diagnosis is mainly clinical, biopsies and cultures, especially fungal cultures, help to confirm diagnosis. Colonies typically appear within 3 to 5 days, but cultures should be held for at least 4 weeks.[2],[3]

Unusual presentation, especially in cases that have failed antibiotics treatment such as the case that we illustrate here, should raise suspicion. Close liaison with other disciplines such as medical microbiology is essential to achieve correct diagnosis so that appropriate treatment can be planned. The fungal culture remains the most specific method in diagnosing sporotrichosis. Currently, there is no reliable serological test for the diagnosis of sporotrichosis.

Treatment of choice for sporotrichosis is oral itraconazole 100-200 mg bis in die (bd) or tds. The recommended period for the antifungal varies from 6 weeks to 3 months. The variation in the period of treatment is likely due to variables of clinical responses following the antifungal treatment.[4],[5],[7] The antifungal therapy is continued even beyond the resolution of lesion as relapse is known to occur. Occasionally, there are cases of sporotrichosis that was resolved spontaneously or resolved with just weekly antifungal.[7] Potassium iodide or Amphotericin B use has been described in the literatures as options.[8] Application of local hot compression or hot packs help as an adjunct as Sporothrix schenckii does not withstand temperatures above 38.5° C.[1],[2],[8]


   Conclusion Top


A proper understanding of the history and clinical manifestation of nodular lymphangitis is required in diagnosing and differentiating this disease from cellulitis so that appropriate treatment can be planned.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Quintal D. Sporotrichosis infectionon mines of the Witwatersrand. J Cutan Med Surg 2000;4:51-4.  Back to cited text no. 1
    
2.
Zhang YQ, Xu XG, Zhang M, Jiang P, Zhou XY, Li ZZ, et al. Sporotrichosis: Clinical and histopathological manifestations. Am J Dermatopathol 2011;33:296-302.  Back to cited text no. 2
    
3.
Bhutia PY, Gurung S, Yegneswaran PP, Pradhan J, Pradhan U, Peggy T, et al. A case series and review of sporotrichosis in Sikkim. J Infect Dev Ctries 2011;5:603-8.  Back to cited text no. 3
    
4.
Hay RJ, Morris-Jones R. Outbreak of sporotrichosis. Curr Opin Infect Dis 2008;21:119-21.  Back to cited text no. 4
    
5.
Tobin EH, Jih WW. Sporotrichoid lymphocutaneous infections: Etiology, diagnosis and therapy. Am Fam Physician 2001;63:326-32.  Back to cited text no. 5
    
6.
Ryncarz RE, Heasley EC, Babinchak TJ. The clinical spectrum of nodular lymphangitis. Hosp Physician 1999;35:63-6.  Back to cited text no. 6
    
7.
Ghodsi SZ, Shams S, Naraghi Z, Daneshpazhooh M, Akhyani M, Arad S, et al. Case report: An unusual case of cutaneous sporotrichosis and its response to weekly fluconazole. Mycoses 2000;43:75-7.  Back to cited text no. 7
    
8.
Giordano CN, Kalb RE, Brass C, Lin L, Helm TN. Nodular lymphangitis: Report of a case with presentation of a diagnostic paradigm. Dermatol Online J 2010;16:1.  Back to cited text no. 8
    

Top
Correspondence Address:
Shaifuzain Ab-Rahman
Department of Orthopedic, Adult Joint Reconstruction Unit, School of Medicine, Universiti Sains Malaysia, Kubang Kerian, Kelantan
Kelantan Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1755-6783.193935

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