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CASE REPORT
Year : 2020  |  Volume : 16  |  Issue : 1  |  Page : 30-32

A surgical surprise reviewed – subcutaneous phaeohyphomycotic cyst


Department of General Surgery, Amrita Institute of Medical Sciences, Kochi, Kerala, India

Date of Submission23-Jan-2020
Date of Acceptance19-Feb-2020
Date of Web Publication07-Jul-2020

Correspondence Address:
Dr. Riju Ramachandran
Department of General Surgery, Amrita Institute of Medical Sciences, Kochi, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/AMJM.AMJM_6_20

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  Abstract 


A 65-year-old female patient presented to the general surgery outpatient department with a nodule in the left palm for 3 months. She gave a history of having pricked her left hand while preparing a composite by crushing dried leaves in her farm. Clinical examination revealed a tender 1 cm × 1 cm hemispherical swelling with well-defined margin in the palm of the left hand which was firm in consistency. There was no foreign body or signs of inflammation. A clinical diagnosis of implantation dermoid cyst was made. The swelling was excised and sent for histopathological examination. Histopathology showed palisaded granuloma with suppuration and cyst formation, with vegetable matter and occasional fungal hyphae resembling a phaeohyphomycotic cyst. A surgically treatable fungal disease may be a differential diagnosis for a swelling in the hands and feet, especially in population exposed to these pathogens.

Keywords: Cyst, fungal disease, hand swellings, phaeohyphomycosis


How to cite this article:
Sivadas N, Ramachandran R, Pillai AV, Krishnan A. A surgical surprise reviewed – subcutaneous phaeohyphomycotic cyst. Amrita J Med 2020;16:30-2

How to cite this URL:
Sivadas N, Ramachandran R, Pillai AV, Krishnan A. A surgical surprise reviewed – subcutaneous phaeohyphomycotic cyst. Amrita J Med [serial online] 2020 [cited 2020 Aug 5];16:30-2. Available from: http://www.ajmonline.org.in/text.asp?2020/16/1/30/289140




  Introduction Top


Subcutaneous phaeohyphomycosis occurs primarily on the extremities, such as the fingers, wrists, knees, or ankles due to the easy exposure of these sites to trauma and contamination of the wounds thus formed by soil, plants, or decomposing wood materials, ultimately leading to direct fungal inoculation. Differentiation of these lesions from fibroma, lipoma, sebaceous cysts, and other disorders is difficult but highly recommended.[1] Surgical excision of the lesion with or without antifungal agents has been widely used in the treatment of subcutaneous phaeohyphomycosis.[2] This case report throws light on the need for considering fungal origin in the backdrop of a traumatic etiology and also the complete response of the condition to surgical treatment without the need for antifungal agents.


  Case Report Top


A 65-year-old female patient presented to our outpatient department with a 1 cm × 1 cm nodule in the left palm for the past 3 months. She gave a history of having pricked her left hand while preparing a composite by crushing dried leaves in her farm. There was delayed healing of the wound. She then noticed a 1 cm × 1 cm swelling over the left palm, insidious in onset, nonprogressive, and associated with a reddish discoloration. There was no history of any discharge from the swelling. She also had a small 1 cm healed wound over the proximal aspect of the right thumb. She attempted some home remedies for her wounds initially, but since there was no satisfactory response, she came to us for further management. She had been treated for renal tuberculosis 40 years ago.

Clinical examination revealed a firm, tender 1 cm × 1 cm hemispherical swelling with well-defined margin in the palm of the left hand. There was a 1 cm linear scar over the dorsal aspect of the proximal third of the right thumb. There was no foreign body or signs of inflammation noticed. A clinical diagnosis of implantation dermoid cyst was made. The swelling was excised and sent for histopathological examination. Prophylactic antibiotics and adequate analgesia were prescribed in the perioperative period. Histopathology showed palisaded granulomas with suppuration and cyst formation with vegetable matter and occasional fungal hyphae resembling a phaeohyphomycotic cyst. Antifungals were not given.

Outcome and follow-up

During a 1-week follow-up, the patient complained of pain over the linear scar of the right thumb. Excision of the scar under local anesthesia was done. At dissection, solid spherical swelling of size 0.5 cm in diameter [Figure 1] was seen under the scar which was sent for histopathological examination and fungal culture considering the previous histopathological report. The histopathological report showed features suggestive of phaeohyphomycotic cyst. The fungal culture report, however, showed no fungal growth. The patient has been free of symptoms after the excision of the swellings on follow-up at 18 months.
Figure 1: Excised specimen

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  Discussion Top


Phaeohyphomycosis is distributed worldwide and is more common in tropical and subtropical climates, being found in soil, wood and decomposing plant debris. They are a group of mycotic infections caused by dematiaceous fungi (pigmented). Phaeohyphomycosis, chromoblastomycosis and eumycotic mycetoma are the major classes of these pigmented fungi.[3] The fungus is present in host tissues as brownish hyphae, pseudohyphae, yeast cells, or combination of these forms.[4] It is a rare infection mostly involving the skin and subcutis, and rarely involving the paranasal sinuses, eyes, central nervous system, lymph nodes, and bone.[5] These opportunistic mycoses are classified into superficial, cutaneous, subcutaneous, systemic, and disseminated forms.

They contain melanin in their cell walls [Figure 2] which increases the virulence of these fungi. Melanin scavenges free radicals produced by phagocytic cells and also prevents hydrolytic enzymes bound to plasma membrane from acting on it. The pathogenic potency of these fungi even in an immunocompetent host is because melanin helps in fungal penetration into the host cell by the formation of fungal appressorium.[6] The immune status of the host thus plays a major role in the clinical presentation.
Figure 2: Histopathology showing the fungal hyphae (brown)

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The clinical presentation ranges from solitary cutaneous nodules to deep subcutaneous abscesses and sometimes nonhealing ulcers or sinuses.[7] The subcutaneous infections are a result of traumatic inoculation and occur on the extremities, fingers, wrists, knees, or ankles.,[7] Histopathological examination and culture are important in the diagnosis of phaeohyphomycosis.

Subcutaneous phaeohyphomycosis is a rare infection; however, the number of cases appears to be increasing in recent years as the number of immunocompromised patients also have increased.[8] Infection usually occurs through traumatic inoculation of the skin and subcutaneous tissue with the contaminated matter.[9],[10] The majority of lesions occur on the feet and legs of outdoor workers, as observed in this patient. The age of the patients ranges from 3 to 60 years.

Males are more commonly affected because of their outdoor occupation.

All dematiaceous fungi are similar in morphology and can be differentiated only by culture.[2] The etiological agents and the anatomic site of involvement usually do not have a bearing on the host reaction to phaeohyphomycosis. Grossly, this condition is characterized by cyst formation with dense collagenous connective tissue and central suppurative necrosis. The overlying epidermis usually looks normal. On microscopy, there is an aggregation of epithelioid histiocytes and numerous giant cells on the wall of the lesion. The presence of pigmented moniliform fungal elements inside the giant cells or extracellularly in the necrotic debris is the pathognomonic feature on microscopy. Fungi measure 2–6 μm wide, with a variable degree of pigmentation and have infrequently branching hyphae. The fungi are closely septate and constricted at their prominent septations.[9]

There appears to be limited information regarding the treatment of subcutaneous phaeohyphomycosis. Surgical excision of subcutaneous lesion is the widely accepted treatment of choice Antifungal therapy is recommended for the recurrence and in immunocompromised patients.[11]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Yoon YA, Park KS, Lee JH, Sung KS, Ki CS, Lee NY. Subcutaneous phaeohyphomycosis caused by Exophiala salmonis. Ann Lab Med 2012;32:438-41.  Back to cited text no. 1
    
2.
Chintagunta S, Arakkal G, Damarla SV, Vodapalli AK. Subcutaneous phaeohyphomycosis in an immunocompetent Individual: A case report. Indian Dermatol Online J 2017;8:29-31.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Rinaldi MG. Phaeohyphomycosis. Dermatol Clin 1996;14:147-53.  Back to cited text no. 3
    
4.
Kwon Chung KJ, Bennett JE. Phaeohyphomycosis. Med Mycology. Pennsylvania: Lea and Febiger; 1992.  Back to cited text no. 4
    
5.
Sharma NL, Mahajan V, Sharma RC, Sharma A. Subcutaneous pheohyphomycosis in India – A case report and review. Int J Dermatol 2002;41:16-20.  Back to cited text no. 5
    
6.
Jacobson ES. Pathogenic roles for fungal melanins. Clin Microbiol Rev 2000;13:708-17.  Back to cited text no. 6
    
7.
Rajendran C, Khaitan BK, Mittal R, Ramam M, Bhardwaj M, Datta KK. Phaeohyphomycosis caused by Exophiala spinifera in India. Med Mycol 2003;41:437-41.  Back to cited text no. 7
    
8.
Mishra D, Singal M, Rodha MS, Subramanian A. Subcutaneous phaeohyphomycosis of foot in an immunocompetent host. J Lab Physicians 2011;3:122-4.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Manoharan M, Shanmugam N, Veeriyan S. A rare case of a subcutaneous phaeomycotic cyst with a brief review of literature. Malays J Med Sci 2011;18:78-81.  Back to cited text no. 9
    
10.
Revankar SG. Phaeohyphomycosis. Infect Dis Clin North Am 2006;20:609-20.  Back to cited text no. 10
    
11.
Allton DR, Parvez N, Ranganath S, Jinadatha C. Surgical management of subcutaneous Colletotrichum gloeosporioides. BMJ Case Rep 2015;2015. pii: bcr2014207540.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]



 

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