|Year : 2014 | Volume
| Issue : 1 | Page : 61-64
Cryptococcal cyst of the acromioclavicular joint: A rare presentation
Ganga S Pilli1, Mahantesh Y Patil2, Mallikarjun S Khanpet3, Tahseen Mujawar3, Lovelesh K Nigam1, Manasi Gosavi1, Sharada Metgud4
1 Department of Pathology, KLE University's J.N. Medical College, Belgaum, Karnataka, India
2 Department of Orthopedics, KLE University's J.N. Medical College, Belgaum, Karnataka, India
3 Department of Medicine, KLE University's J.N. Medical College, Belgaum, Karnataka, India
4 Department of Microbiology, KLE University's J.N. Medical College, Belgaum, Karnataka, India
|Date of Web Publication||2-Jul-2014|
Ganga S Pilli
Department of Pathology, KLE University's J.N. Medical College, Belgaum - 590 010, Karnataka
Source of Support: None, Conflict of Interest: None
Cryptococcus neoformans is yeast and it commonly affects immuno-compromised patients, including AIDS. Most infections with C. neoformans consist of a lung lesions. However, meningitis or meningoencephalitis and localized infections can occur as part of systemic infection. Case: A 32-year-old patient, presented with a swelling over the right shoulder with pain and restriction of movements. The pain has increased in the past 10 days. Patient has a history of hypertension. He developed end stage renal disease and was put on dialysis. He was advised to undergo renal transplant. Renal transplant was done a year back. O/E patient was moderately built, but poorly nourished and pallor was present. On local examination, swelling was seen over the anterior aspect of right shoulder and right acromio-clavicular region with diffuse margins and tenderness. On fine-needle aspiration cytology (FNAC), the smears revealed yeasts of Cryptococci, which were confirmed with special stains and culture. With the diagnosis of cryptococcal cyst, patient underwent surgical excision and was put on anti-fungal therapy and is relieved of his symptoms. The present case is a rare presentation of Cryptococci in an immune-compromised patient of renal transplant occurring in an unusual site. In conclusion, a rare case of cryptococcal cyst of acromio-clavicular joint could be diagnosed by FNAC.
Keywords: Cryptococci, cyst, immuno-compromised state
|How to cite this article:|
Pilli GS, Patil MY, Khanpet MS, Mujawar T, Nigam LK, Gosavi M, Metgud S. Cryptococcal cyst of the acromioclavicular joint: A rare presentation. Indian J Health Sci Biomed Res 2014;7:61-4
|How to cite this URL:|
Pilli GS, Patil MY, Khanpet MS, Mujawar T, Nigam LK, Gosavi M, Metgud S. Cryptococcal cyst of the acromioclavicular joint: A rare presentation. Indian J Health Sci Biomed Res [serial online] 2014 [cited 2020 Jan 27];7:61-4. Available from: http://www.ijournalhs.org/text.asp?2014/7/1/61/135084
| Introduction|| |
Cryptococcus is ubiquitous encapsulated fungi which was first isolated from peach juice and described by Sanfelice in 1894.  Cryptococcus is found in avian feces, most commonly pigeon droppings, but also exists in soil, spoiled milk, fruits, and other food products. It can cause disease in immune-competent as well as in immuno-compromised hosts.
The latter group with, particularly those with cell mediated immune defects are at the greatest risk for severe disseminated disease.  Cryptococcal infection most commonly affects lungs and the respiratory tract and is believed to be the main portal of entry. The resultant pulmonary disease is often subclinical and transient. Hematogenous dissemination may infect any organ, but has a preference for central nervous system (CNS) with meningoencephelitis as the most common clinical manifestation. Rarely skin, kidneys, bone etc., can be involved. Literature search has revealed only 22 documented cases of cryptococcal arthritis, tendinitis and tenosynovitis.  Here, we present a rare case of Cryptococcus presenting as a cystic mass over the anterior surface of acromio-clavicular joint and the case is presented for its rare presentation in a patient who was immuno-compromised because of renal transplantation. This is the first documented case of Cryptococcus presenting as a cyst involving the synovium on the extra articular surface of the acromio-clavicular joint, which could be excised and treated with anti-fungal therapy.
| Case Report|| |
A 32-year-old male patient presented with a history of swelling over the right shoulder since 5 months, which gradually increased to the present size and is associated with pain and restriction of movements. The pain has increased over the period of last 10 days.
In the past history, patient was a known case of hypertension since last 10 years and he was on treatment.
Patient developed end stage renal disease and was put on dialysis since past 4 years. He was advised to undergo renal transplant. Renal transplant was done a year back and there were no posttransplantation complications or any illness until last 5 months. The patient was on drugs: Vingraf, wysolone, azoran and atenolol.
On examination, patient was moderately built but poorly nourished. Patient was anemic. Systemic examination of cardiovascular system, CNS, respiratory system and PA were unremarkable.
On local examination
The swelling was seen over the anterior aspect of right shoulder and right acromio-clavicular joint region. The swelling was 4 × 3 cm, had diffuse margins and tenderness was present [Figure 1].
Except for the hemoglobin of 10 g% and normocytic normochromic anemia, all the other investigations were within the normal range, including renal parameters.
Magnetic resonance imaging of the shoulder revealed a cystic lesion over the anterior aspect of right acromio-clavicular joint involving the synovium on the extra articular surface without involving the cavity of shoulder joint [Figure 2].
|Figure 2: Magnetic resonance imaging of the shoulder to show cystic swelling over the anterior aspect of the acromio-clavicular joint|
Click here to view
Fine-needle aspiration cytology of the mass was done. To our surprise, revealed yeasts of Cryptococcus on May Grunwald Giemsa stain [Figure 3] which were confirmed with special stain [Figure 4] and culture yielded characteristic smooth, mucoid and cream colored colonies of Cryptococcus neoformans on Sabouraud's dextrose agar [Figure 5].
|Figure 3: Microphotograph to show yeasts of Cryptococci (May Grunwald Giemsa, ×450)|
Click here to view
|Figure 4: Microphotograph to show yeasts of Cryptococci (Periodic acid Schiff's, ×200)|
Click here to view
|Figure 5: Smooth, mucoid and cream colored colonies of Cryptococci on Sabouraud's dextrose agar|
Click here to view
The patient underwent surgical excision of the cyst and was put on anti-fungal therapy. He is relieved of the symptoms. After 6 months of follow-up, patient is comfortable with his routine work and has no complaints.
| Discussion|| |
Cryptococcus gains entry into the lungs through respiratory passage and remain dormant depending on the host immune reaction. A normally functioning host immune system is capable of eliminating the infection at this stage. However, it proliferates, if the host's immune system is compromised, as it occurs in patients with HIV or in patients receiving immunosuppressive agents in renal transplant patients. It can disseminate or can be localized to certain organs. 
Cryptococcal infection can be asymptomatic, chronic or acute. Pulmonary infection may spread systemically to CNS. Pulmonary infection, in most instances is asymptomatic. Pneumonia and acute respiratory distress syndrome have been reported in immuno-compromised patients.  Cryptococcosis of the CNS is life threatening and presents as meningitis and meningoencephelitis. Cryptococcal meningitis generally presents with headache of insidious onset. Fever usually do not occur until late in the disease course and nuchal rigidity is usually absent. 
Cryptococcal infection is the leading cause of death in HIV infected patients with an incidence of 30% and mortality of 30-60%. The mortality in transplant patients is higher (20-100%). 
Less commonly, infection can be localized to other organs. In literature, there is documentation of 22 case of Cryptococcus involving the joints. One of the cases presented with tendinitis, tenosynovitis and carpal tunnel syndrome. Immunosuppression due to underlying disease or immunosuppressive therapy appeared to be the major cause for involvement of the joints. Five of the patients had undergone renal transplant and one had undergone liver transplant. In other cases, the underlying diseases were AIDs, sarcoidosis, diabetes mellitus, systemic lupus erythematosis and in few cases no underlying cause was detected.  Disseminated disease was more common than isolated joint involvement. Only four patients presented with solely joint involvement. Of the 22 cases, six patients died and rest were cured with anti-fungal therapy.
Detection of cryptococcal antigen in serum and cerebrospinal fluid using latex agglutination test is more promising than antibody detection. The Cryptococci can be stained with negative staining with India ink or stained with special stains such as periodic acid Schiff's (PAS), Gomori's silver methenamine, alcian blue and Mayer's mucicarmine stains.
Patient can be treated with Intravenous fluconozole (600 mg/day) and 5-fluorocytosine (1500 mg every 6 th hourly) for 2 weeks followed by oral fluconozole (400 mg/day) for 8-10 weeks. Amphoterisin B and 5-fluorocytosine combination also can be used.
To the best of our knowledge, the present case is the first documented case of Cryptococcus presenting as a cyst involving the synovium on the extra articular surface of the acromio-clavicular joint, which could be excised and treated with anti-fungal therapy. Immunosuppression or immuno-suppressive agents must be kept in mind for diagnosing this disease. Early detection of the opportunistic infection and immediate specific treatment could be lifesaving as in the present case.
| References|| |
|1.||Sanfelice F. Contributo alla morfologia e biologica dei blastomiceti che si sviluppano nei succhi di alcuni frutti. Ann Ig 1894;4:463. |
|2.||Henderson HM, Chapman SW. Infections due to fungi, actinomyces and nocardia. In: Reese RE, Betts RF, editors. A Practical Approach to Infectious Disease. 4 th ed. Boston: Little, Brown and Company; 1996. p. 672-5. |
|3.||Bruno KM, Farhoomand L, Libman BS, Pappas CN, Landry FJ. Cryptococcal arthritis, tendinitis, tenosynovitis, and carpal tunnel syndrome: Report of a case and review of the literature. Arthritis Rheum 2002;47:104-8. |
|4.||Nadrous HF, Antonios VS, Terrell CL, Ryu JH. Pulmonary cryptococcosis in nonimmunocompromised patients. Chest 2003;124:2143-7. |
|5.||Hajjeh RA, Brandt ME, Pinner RW. Emergence of cryptococcal disease: Epidemiologic perspectives 100 years after its discovery. Epidemiol Rev 1995;17:303-20. |
|6.||Mitchell TG, Perfect JR. Cryptococcosis in the era of AIDS-100 years after the discovery of Cryptococcus neoformans. Clin Microbiol Rev 1995;8:515-48. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]