Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 14  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
E-IJD® - CASE REPORT
Year : 2022  |  Volume : 67  |  Issue : 3  |  Page : 314
A rare case of zoonotic infection in human – Emphasising the one health concept


Department of Dermatology, Calcutta National Medical College, Kolkata, West Bengal, India

Date of Web Publication22-Sep-2022

Correspondence Address:
Abhishek De
Flat Number 3A Arcadia 1 Dream Park Sonarpur Station Road, Kolkata - 700 103, West Bengal
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_102_20

Rights and Permissions

   Abstract 


The One Health concept recognizes that the health of people is connected to the health of animals and the environment. A 21-year-old female patient presented with several nodular and cystic swellings on the front and sides of her neck for the past 2 years. Histopathology showed sulfur granules surrounded by neutrophils in an area of granulation tissue with filamentous Gram-negative bacilli in the granules. She was a regular cattle handler since her childhood, and one of her cows on the farm had died from a similar sort of illness 2 years back at the same period when her disease began. We made a provisional diagnosis of actinobacillus infection and started the patient on doxycycline 100 mg twice daily. With this treatment, the patient showed an excellent clinical response and her condition healed with resultant scarring within 7 weeks of treatment.


Keywords: Actinobacillus, one health concept, zoonotic infection


How to cite this article:
Dutta A, De A, Das S, Kar C, Halder D. A rare case of zoonotic infection in human – Emphasising the one health concept. Indian J Dermatol 2022;67:314

How to cite this URL:
Dutta A, De A, Das S, Kar C, Halder D. A rare case of zoonotic infection in human – Emphasising the one health concept. Indian J Dermatol [serial online] 2022 [cited 2022 Sep 30];67:314. Available from: https://www.e-ijd.org/text.asp?2022/67/3/314/356723





   Introduction Top


The One Health concept recognizes that the health of people is connected to the health of animals and the environment. It is a multifactorial and transdisciplinary approach with the goal of achieving optimal health outcomes recognizing the interconnection between people, animals, and the environment. One health concept has become very important in the recent years because these changes have led to the emergence and reemergence of many diseases. People who have closer contact with a large number of animals such as farmers, abattoir workers, zoo/pet-shop workers, and veterinarians are at higher risk of contracting a zoonotic disease.[1] We present a case to emphasize how the awareness of “one health concept” helps in the diagnosis and management of a puzzling case.


   Case Summary Top


A 21-year-old female patient presented to the Dermatology department of our hospital with complaints of several nodular and cystic swellings on the front and sides of her neck for the past 2 years [Figure 1].
Figure 1: Several nodular and cystic swellings in the front of the neck region

Click here to view


The patient was apparently well 2 years back since she noticed a small nodular swelling in the front of neck region which had increased to present size with some lesions in the right forearm in a sporotrichoid distribution 8–9 months back which had now healed with hyperpigmentation [Figure 2]. The patient was prescribed a 6-month Category-I regimen of antitubercular drugs by a local doctor after making a diagnosis of scrofuloderma and was also given multiple courses of antibiotics from another health care center without any clinical response.
Figure 2: Lesions in the right forearm in a sporotrichoid distribution

Click here to view


Fine needle aspiration from neck swelling showed plenty of histiocytes in clusters, fibrocapillary tissue fragments, macrophages, degenerated neutrophils, and lymphocytes with Gram-staining from the smear material revealing Gram-negative bacilli. The mandibular X-ray (Occipitomental and Lateral view) was normal.

Skin lesion for histopathologic examination showed sulfur granules surrounded by neutrophils in an area of granulation tissue [Figure 3] and [Figure 4] with Gram stain revealing filamentous Gram-negative bacilli in the granules and also in the fluid sent for microbiological evaluation [Figure 5]a and [Figure 5]b. Ziehl-Neelsen stain for acid-fast bacillus (1%), standard bacterial culture, and fungal culture report were negative. Gomori methanamine staining (GMS) and periodic acid-Schiff stain (PAS) [Figure 6]a and [Figure 6]b revealed no fungal hyphae in the granules.
Figure 3: Histopathology 10 × view showing sulfur granules surrounded by neutrophils in an area of granulation tissue

Click here to view
Figure 4: Histopathology 40 × view showing an enlarged section of Sulphur granules

Click here to view
Figure 5: (a and b) Gram-stain of the organism showing filamentous Gram-negative bacilli

Click here to view
Figure 6: GMS and PAS stain of the organism was done which was negative

Click here to view


The site of infection and presence of sulfur granules implied actinomycosis infection; however, noninvolvement of jawbones and presence of Gram-negative bacilli in both. Fine needle aspiration and histopathology created a diagnostic dilemma. Ziehl-Neelsen stain for acid-fast bacillus (1%) being negative, we ruled out nocardiosis.

Our other differentials including scrofuloderma, deep fungal infection considering the sporotrichoid spread, and lymhangiosarcoma could be excluded by the investigative findings.

In the meantime, on again questioning the patient about the mode of onset of the disease, she give a history that she had a small farm in her house and a regular cattle handler since her childhood. She also gave a history that one of her cows on the farm had died from a similar sort of illness 2 years back at the same period when her disease began.

With this background and after an extensive review of literature, we made a provisional diagnosis of actinobacillus infection and started the patient on doxycycline 100 mg twice daily. With this treatment, the patient showed excellent clinical response, and her condition healed with resultant scarring within 7 weeks of treatment. [Figure 7] and [Figure 8] The patient was discharged in good general health, and she is on follow up for the last 6 months in our outpatient department, without any relapse or recurrence of her condition.
Figure 7: Lesions in the neck regressing after 5 weeks of treatment

Click here to view
Figure 8: Excellent clinical response after 7 weeks of treatment

Click here to view



   Discussion Top


Actinobacillosis is a chronic infectious self-limiting disease caused most commonly by Gram-negative cocobacilli Actinobacillus ligniersii. It is commonly responsible for the wooden tongue disease characterized by the presence of granulomas with pus containing small, hard yellow to white granules. It affects mostly cattle and sheep but is also known to occur in horses, pigs, and dogs.[2]

After an extensive review of the literature, we found that there had been only nine cases of Actinobacillus infection in man, three of them fatal.[3]

There have been reports of actinobacillar meningitis by Orda and WiZnitzer (1911) complicating otitis and mastoiditis.[3] Additional reports include fatal meningitis in a newborn infected by cow's milk, sepsis developing in a butcher with a traumatic abdominal wound probably produced by a contaminated knife; fulminating suppurative bronchopneumonia with metastatic abscesses in the liver, kidneys, and spleen, fatal mitral endocarditis with focal embolic nephritis and embolic disturbances in the spleen, the meninges and the skin;[3] ileocecal lymph node involvement in a girl operated with a diagnosis of acute appendicitis (Flamm and Jonas 1956),[4] conjunctivitis (Flamm and Wiedermann 1962),[5] and pneumonia (Pavckova et al. 1973).[6]

Actinobacillosis was first described by Lignieres and Spitz (1902) in cattle in Argentina and was subsequently reported in the sheep and dog.[7] The appearance of multiple nodules in the subcutaneous tissue is a common feature in infected animals. The mouth, tongue, pharynx, internal organs, and regional lymph nodes are frequently involved. The nodules become necrotic and ulcerated, discharging a yellow-green viscous purulent content.[7] Thompson had reported for the first time, the existence of actinobacillosis is a common malady of cattle in the United States. He concluded that “Actinobacillosis is common among cattle in the United States. It would seem that the condition here is similar to that in other countries where the greater percentage of so-called bovine actinomycosis is due to Actinobacillus lignieresi.”[7]

In humans, Actinobacillus is usually transmitted from cattle and sheep.[8] There have been two reports of serious facial infection after a horse bite, from which small Gram-negative rod Actinobacillus lignieresii was isolated.[9],[10] Contrary to actinomycetes which demonstrates a predilection for the skeletal system, the Actinobacillus has a tendency to invade the soft tissue and organs like the lungs, abdominal and pelvic viscera, and lymph nodes (Custis et al. 1944) [Table 1].[11],[12]
Table 1: Difference between ACTINOBACILLOSIS and ACTINOMYCOSIS

Click here to view


In one fatal case of actinobacillosis, a pathological study was made. Sections of the lung, liver, spleen, and left kidney were studied which consist of focal, sharply circumscribed zones of exudation with usually advanced necrosis of the exudate centrally and granulomatous proliferative changes peripherally. Early lesions are composed almost entirely of polymorphonuclear neutrophilic leukocytes without necrosis or peripheral granulomatous reaction.[7]

The present case of Actinobacillus infection had a chronic and long evolution. The contact of the patient with cows and goats might lead us to suspect the exogenic origin of the infection, either through a skin wound or by indirect contamination as she had given a history of the death of one of her cows at the same period of time when her disease began.

We report this case because of the extreme rarity of this case as we believe this is the first reported case from Asia. Moreover, this case taught us to understand the one health concept and the importance of physicians' awareness about zoonotic infection in susceptible hosts.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Cantas L, Suer K. Review: The important bacterial zoonoses in “one health” concept. Front Public Health 2014;2:144.  Back to cited text no. 1
    
2.
Thompson L, Willius FA. Actinobacillus bacteremia. JAMA 1932;99:298-300.  Back to cited text no. 2
    
3.
Orda R, WiZnitzer T. Actinobacillus ligniersii human infection. J R Soc Med 1980;73:295-7.  Back to cited text no. 3
    
4.
Flamm H, Jonas R. Actinobaccilosis in a child. Wien Klin Wochenschr 1956;68:671-3.  Back to cited text no. 4
    
5.
Flamm H, Wiedermann G. Infections caused by Actinobacillus lignieresi in man. Z Hyg Infektionskr 1962;148:368-74.  Back to cited text no. 5
    
6.
Block PJ, Yoran C, Fox AC, Kaltman AJ. Actinobacillus actinomycetemcomitans endocarditis: Report of a case and review of the literature. Am J Med Sci 1973;266:387-92.  Back to cited text no. 6
    
7.
Beaver DC, Thompson L. Actinobacillosis of man: Report of a fatal case. Am J Pathol 1933;9:603-22.1.  Back to cited text no. 7
    
8.
Peel MM, Hornidge KA, Luppino M, Stacpoole AM, Weaver RE. Actinobacillus spp. and related bacteria in infected wounds of humans bitten by horses and sheep. J Clin Microbiol 1991;29:2535-8.  Back to cited text no. 8
    
9.
Benaoudia F, Escande F, Simonet M. Infection due to Actinobacillus lignieresii after a horse bite. Eur J Clin Microbiol Infect Dis 1994;13:439-40.  Back to cited text no. 9
    
10.
Dibb WL, Digranes A, Tønjum S. Actinobacillus lignieresii infection after a horse bite. Br Med J (Clin Res Ed) 1981;283:583-4.  Back to cited text no. 10
    
11.
Custis DL, Halley H, Bacon CM. Actino-bacillus lignièresi endocarditis. Arch Pathol 1944;38:332-6.  Back to cited text no. 11
    
12.
Paucková V, Lásková H, Krakovic B. Actinobacillus lignieresii human infection. Zentralbl Bakteriol Orig A 1973;224:489-91.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (2,845 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Summary
   Discussion
    References
    Article Figures
    Article Tables

 Article Access Statistics
    Viewed146    
    Printed2    
    Emailed0    
    PDF Downloaded38    
    Comments [Add]    

Recommend this journal