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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 2  |  Page : 55-57

Disseminated rhinosporidiosis mimicking pulmonary metastasis – A case report


1 Department of Respiratory Medicine, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala, India
2 Department of ENT, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala, India

Date of Submission29-Jan-2021
Date of Decision21-Apr-2021
Date of Acceptance22-Apr-2021
Date of Web Publication21-Jun-2021

Correspondence Address:
Dr. Rohith Satheesh
Consultant, Department of Respiratory Medicine, Kerala Institute of Medical Sciences, Anayara P.O, Trivandrum – 695 029, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jalh.jalh_1_21

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  Abstract 


Rhinosporidiosis is a granulomatous disease caused by Rhinosporidium seeberi, mostly affecting the nasal mucosa. Extranasal involvement is also reported involving eyes, oral cavity, and skin. Herein, we present a rare case of a patient with disseminated rhinosporidiosis who presented with pulmonary lesions which resembled metastasis.

Keywords: Disseminated, lung involvement, rhinosporidiosis


How to cite this article:
Balakrishnan R, Satheesh R, Felix V, Padmanabhan A. Disseminated rhinosporidiosis mimicking pulmonary metastasis – A case report. J Adv Lung Health 2021;1:55-7

How to cite this URL:
Balakrishnan R, Satheesh R, Felix V, Padmanabhan A. Disseminated rhinosporidiosis mimicking pulmonary metastasis – A case report. J Adv Lung Health [serial online] 2021 [cited 2021 Jul 28];1:55-7. Available from: http://www.jalh.com/text.asp?2021/1/2/55/318905




  Introduction Top


Rhinosporidiosis is a granulomatous disease endemic in Asia, Africa, and Europe, with the highest prevalence in Southern India and Sri Lanka. It is generally known to involve the upper respiratory tract. Pulmonary involvement is rare and is mostly limited to the trachea.

We present a rare case of disseminated rhinosporidiosis, in a patient with diabetes mellitus, who presented with pulmonary lesions which were suggestive of metastatic malignancy.


  Case Report Top


A 68-year-old gentleman, diabetic on oral hypoglycemic agents, presented to the ear, nose, and throat department with complaints of multiple swellings on the face. He had no respiratory or constitutional symptoms. He was a smoker but had stopped smoking 15 days before presentation. His smoking index was 192. He had undergone surgical excision of similar swellings over face, eyes, and thigh seven times within the last 10 years, all of which were proved to be rhinosporidiosis on histopathological examination. He was advised to take tablet dapsone 100 mg once daily, which he took irregularly. On examination, two swellings were noted on the face– a 2 cm × 2 cm swelling over the right eyelid extending till the lateral canthus and a 0.5 cm pinkish swelling inside the nasal cavity, both being nontender, firm, and mobile. Healed scars were noted over the left cheek, right lower limb, and right thigh. General and systemic examination was unremarkable. Video laryngoscopy revealed a 1 cm × 1 cm pinkish-red proliferative mass arising from the right pharyngeal wall.

Blood investigations showed hemoglobin 15.5 g/dL, total white blood cell count 9100/mm3 (P59%L28%M9%E2%), platelet count 2.4 L/mm3, random blood glucose 123 mg/dL, and HbA1C 6.8%. Liver function test and renal function test were within normal limits. Infectious virology screening including HIV was also negative.

The otorhinolaryngologist decided to proceed with surgical excision of the swellings, and as a part of preoperative evaluation, the patient was referred to the respiratory medicine department. Routine chest X-ray revealed multiple rounded opacities of varying sizes, involving bilateral lower zones, mid zones, and retrocardiac area [Figure 1]. Computed tomography of the chest showed well-defined mass lesions in the mid and lower zones of both lungs [Figure 2]. The largest one was in the right middle lobe measuring 5.5 cm × 4.3 cm. There was a soft tissue density lesion on the lateral aspect of the trachea measuring 9.5 mm × 7 mm.
Figure 1: Chest X-ray posteroanterior view demonstrating multiple rounded opacities of varying sizes, involving bilateral lower zones, mid zones including retrocardiac area

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Figure 2: Axial computed tomography of the chest demonstrating well-defined mass lesions in bilateral lower zones, largest one in the middle lobe measuring 5.5 cm × 4.3 cm

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The patient being an elderly smoker with pulmonary mass lesions, we considered malignancy as the first diagnosis, followed by fungal nodules and noninfectious causes such as vasculitis as the differential diagnosis.

We proceeded with ultrasound-guided fine-needle aspiration cytology of the pulmonary lesion, which showed granuloma and foreign body giant cells along with fungal sporangia containing spores, which was consistent with rhinosporidiosis [Figure 3].
Figure 3: Fine-needle aspiration cytology demonstrating sheets and collections of histiocytes forming granuloma, admixed with lymphocytes, foreign body giant cells, and fungal sporangia containing spores

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Surgical excision was done for swellings over the eyelid, nasal cavity, and the pharynx, all of which were proved to be rhinosporidiosis on histopathological examination.


  Discussion Top


Rhinosporidiosis is endemic in Asia, Africa, and Europe, with the highest prevalence in Southern India and Sri Lanka.[1] The causative agent was first identified in 1890 and was named as Coccidium seeberia.[2] Its lifecycle was described by Seeber in 1900.[2] It was thought to be a water mold till Herr et al. established similarities with a group of fish parasites known as the DRIP clade and included in this group, which was renamed as Mesomycetozoa.[3] Commonly described mode of transmission is direct contact with spores of Rhinosporidium seeberi through exposure to stagnant water sources by which the spores enter the body via traumatized epithelium. Hence, the disease is common in paddy cultivators and river sand workers.[4] It is also commonly seen in swimmers and people who take bath in ponds whose waters are contaminated by this organism. Less common modes include dust and infected clothing.[5] Once infected, local spread of disease occurs by autoinoculation of endospores.[4]

The treatment of choice is wide local excision of the lesions with electrocoagulation of the base. Antifungals such as griseofulvin and amphotericin B and antibiotics such as trimethoprim-sulfadiazine were proposed as adjuvant medical treatment, and all these are endospore-static. Later studies focused on dapsone, a bacteriostatic sulfonamide, which can arrest the maturation of sporangia and promote fibrosis in the stroma. It can also prevent colonization and infection of new sites after the release of endospores from surgically traumatized polyps.[6]

In our case, the patient was an adult male from the urban area of Thiruvananthapuram, who worked as a manager in an electronics company. He gives a history of travel all over India throughout his career but denies the habit of swimming in ponds. We guess that he might have had exposure to stagnant water sources unknowingly or else there could be entry of spores into traumatized skin through dust. It is said that spores from nasopharynx may spill over into the mucosa of larynx and trachea during episodes of bleeding or surgery.[7] However, the extensive cutaneous and visceral involvement, in this case, points to hematogenous spread. As dapsone has a modest benefit in delaying disease progression,[8] he was reinitiated on tablet dapsone 100 mg once daily and advised to be on a regular follow-up.

Though rhinosporidiosis is known to recur even after surgical treatment, the common clinical forms are nasopharyngeal lesions followed by ocular and cutaneous lesions. A few cases of tracheobronchial rhinosporidiosis have been reported from India. Our case becomes unique because of extensive pulmonary involvement, which is rarely encountered in clinical practice. Rajakannu et al. have reported a case of disseminated rhinosporidiosis with pulmonary involvement and is very similar to our case.[9] This case is being reported because of its rarity. The presentation as lung masses would tend to misguide one to consider metastatic disease as the first possibility, especially in the backdrop of his smoking history. To the best of our knowledge, only two cases of disseminated rhinosporidiosis with hematogenous spread involving multiple viscera have been reported to date, both were in the 1950s, and this case report is intended to remind the medical community that disseminated rhinosporidiosis can occur even in this era.[10]

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.

Acknowledgement

We would like to acknowledge the contributions of. Dr. Leena Devi.K.R, Dr. Rachel Abraham, Dr. Ann Susan Robert, Dr.Nair Parvati Ramachandran and Dr. Rakhi M.R, Department of Pathology who were involved in Cytological and Histopathological examination.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Fredricks DN, Jolley JA, Lepp PW, Kosek JC, Relman DA. Rhinosporidium seeberi: A human pathogen from a novel group of aquatic protistan parasites. Emerg Infect Dis 2000;6:273-82.  Back to cited text no. 1
    
2.
Das S, Kashyap B, Barua M, Gupta N, Saha R, Vaid L, et al. Nasal rhinosporidiosis in humans: New interpretations and a review of the literature of this enigmatic disease. Med Mycol 2011;49:311-5.  Back to cited text no. 2
    
3.
Herr RA, Ajello L, Taylor JW, Arseculeratne SN, Mendoza L. Phylogenetic analysis of Rhinosporidium seeberi's 18S small-subunit ribosomal DNA groups this pathogen among members of the protoctistan Mesomycetozoa clade. J Clin Microbiol 1999;37:2750-4.  Back to cited text no. 3
    
4.
Arseculeratne SN. Recent advances in rhinosporidiosis and Rhinosporidium seeberi. Indian J Med Microbiol 2002;20:119-31.  Back to cited text no. 4
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5.
Thappa DM, Venkatesan S, Sirka CS, Jaisankar TJ, Gopalkrishnan, Ratnakar C. Disseminated cutaneous rhinosporidiosis. J Dermatol 1998;25:527-32.  Back to cited text no. 5
    
6.
Arseculeratne SN. Chemotherapy of rhinosporidiosis: A review. J Infect Dis Antimicrob Agents 2009;26:21-7.  Back to cited text no. 6
    
7.
Madana J, Yolmo D, Gopalakrishnan S, Saxena SK. Rhinosporidiosis of the upper airways and trachea. J Laryngol Otol 2010;124:1139-41.  Back to cited text no. 7
    
8.
Job A, Venkateswaran S, Mathan M, Krishnaswami H, Raman R. Medical therapy of rhinosporidiosis with dapsone. J Laryngol Otol 1993;107:809-12.  Back to cited text no. 8
    
9.
Rajakannu M, Sri Vengadesh G, Pai D, Jagdish S. Disseminated rhinosporidiosis – An unusual presentation with pulmonary involvement. Int J Dermatol 2006;45:297-8.  Back to cited text no. 9
    
10.
Rajam RV, Viswanathan GC, Rao AR. Rhinosporidiosis: A study with a report of a fatal case with systemic dissemination. Indian J Surg 1955;17:269-98.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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