SCIENTIFIC ARTICLE

Candida parapsilosis Tenosynovitis in an Immunocompetent Patient: Case Report and Review of Literature Hetal Fichadia, MD, Charles Layman, MD

We describe a case of fungal tenosynovitis with Candida parapsilosis, which is an uncommonly reported agent causing tenosynovitis. It occurred in an immunocompetent individual, and the patient underwent an extensive noninfectious work-up for ongoing swelling and stiffness before being correctly diagnosed and treated. We emphasize the importance of considering atypical infections in the differential diagnoses in a patient presenting with indolent symptoms of tenosynovitis. (J Hand Surg Am. 2015;40(5):993e996. Copyright Ó 2015 by the American Society for Surgery of the Hand. All rights reserved.) Key words Atypical infections, Candida parapsilosis, fungal.

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with fungi or mycobacteria are usually seen in immunocompromised individuals. In such patients, these infections are often in the differential in the appropriate clinical setting. However, deep fungal infection is rare in immunocompetent patients. We describe a case of fungal tenosynovitis in an immunocompetent patient presenting with chronic stiffness, mild swelling, and reduced range of motion instead of the usual infectious signs such as acute pain and swelling. EEP SPACE ATYPICAL HAND INFECTIONS

CLINICAL CASE A 46-year-old right-handed man was referred to our hand practice for swelling and reduced range of motion of his left middle finger. He had a workrelated crush injury (with intact skin) from heavy From the Division of Plastic and Reconstructive Surgery, Oregon Health and Sciences University; and Hand and Microsurgery Associates Northwest, Providence St. Vincent Medical Center, Portland, OR. Received for publication November 7, 2014; accepted in revised form January 20, 2015. No benefits in any form have been received or will be received related directly or indirectly to the subject of this article. Corresponding author: Hetal Fichadia, MD, Division of Plastic and Reconstructive Surgery, Oregon Health and Sciences University, 3181 SW Sam Jackson Park Rd., Mailing Code L352A, Portland, OR 97239; e-mail: [email protected]. 0363-5023/15/4005-0020$36.00/0 http://dx.doi.org/10.1016/j.jhsa.2015.01.021

machinery to his left middle finger almost 3 years previously. This was initially treated with nonsteroidal anti-inflammatory medications. Owing to persistent pain and swelling, a diagnosis of a traumatic ganglion cyst less than one cm in diameter on the flexor tendon sheath in the proximal aspect of left long finger was made. This was excised 2 months after the injury through a proximal digital crease incision. Following this excision, he had drainage from the incision, swelling, and decreased range of motion. Cultures of the drainage were negative, and the drainage stopped in one week. He was treated with antibiotics (cephalexin, ciprofloxacin) and hand therapy. He also received a total of 5 steroid injections at subsequent visits (10 mg of triamcinolone, 2e6 wk apart) after the drainage had stopped. He never experienced complete symptomatic relief but reported temporary improvement after steroid injections and continued to work. Twenty-five months after injury, he returned with decreased range of motion and was found to have a nodule in the A2-A3 pulley area (as described in outside records). The nodule appeared to be in the same location as the prior cyst. He had that excised at an outside hospital shortly thereafter. Two volar incisions were made—one oriented obliquely over the volar proximal phalanx and another over the proximal digital crease. Intraoperative examination showed a “pumpkin seede like” inflammatory nodule. This was sent to pathology

Ó 2015 ASSH

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Published by Elsevier, Inc. All rights reserved.

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CASE REPORT OF CANDIDA PARAPSILOSIS INFECTION

FIGURE 2: Pathology slide of tenosynovium from the left middle finger shows a subepithelial mixed inflammatory reaction with predominance of histocytes, associated with a scar. (Hematoxylineosin stain; magnification  40.)

and bilateral ulnar nerve transpositions, all of which he recovered from uneventfully. He did not have diabetes mellitus. Physical examination revealed swelling and motion at the metacarpophalangeal joint 0/60, proximal interphalangeal joint 30/60, and distal interphalangeal joint 0/40. Motion in the other digits of both hands was normal. Two-point discrimination was 5 mm in all digits. Grip strength was 52 kgf on the right and 28 kgf on the left. There was moderate swelling and mild tenderness diffusely over the volar aspect of the proximal and middle phalanges of the left middle finger. He underwent left middle finger flexor tenosynovectomy through a Bruner incision extending from the distal palmar crease to just distal to the distal digital flexion crease 35 months following his original injury. Intraoperative findings were consistent with marked tenosynovitis extending along the length of the flexor tendon in the finger. Tenosynovium was sent for pathology (in formalin) and cultured for bacteria, fungi, and acid-fast bacilli. The pathology report was consistent with chronic tenosynovitis. The culture was positive for Candida parapsilosis sensitive to amphotericin and voriconazole. After receiving this microbiological diagnosis, we requested our pathologist to stain the tissue with fungus-specific stain (methanamine silver stain), which showed the fungal elements that were previously not seen with hematoxylin-eosin stain (Figs. 2e4). An infectious disease consult provided subsequent management. Immune markers including CD4 count, immunoglobulin G (IgG) and IgM were within normal

FIGURE 1: Magnetic resonance imaging shows fluid within the tendon sheath surrounding the flexor digitorum superficialis and flexor digitorum profundus tendons at the level of the third metacarpal and third proximal phalanx and edema within the soft tissues volar to the third metacarpal and third proximal phalanx.

but not microbiology. Histopathological examination (hematoxylin-eosin stain) showed fibromembranous tissue and abundant fibrinous debris with degeneration—reported as consistent with ganglion cyst. A second pathologist from a different institution reviewed and confirmed the findings. The patient continued to have swelling, stiffness, and mild pain in the left middle finger after surgery. Further work-up included serology for rheumatoid arthritis (rheumatoid arthritis factor slightly elevated, human leukocyte antigen B27 positive, erythrocyte sedimentation rate/C-reactive protein normal, antinuclear antibodies negative) and magnetic resonance imaging, which showed flexor tendon tenosynovitis of the left middle finger at the level of the metacarpophalangeal joint and proximal phalanx (Fig. 1). A rheumatologist concluded that rheumatoid arthritis was unlikely and recommended evaluation by a hand surgeon, and the patient came to us. Past medical history included several trigger finger releases (including of the left middle finger in the distant past), bilateral carpal tunnel decompressions, J Hand Surg Am.

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FIGURE 3: Pathology slide of tenosynovium from the left middle finger with focus on the region where fungal elements are identified in the next slide. (Hematoxylin-eosin stain;  100.)

FIGURE 4: Pathology slide of tenosynovium from the left middle finger. This fungus-specific stain highlights fungal elements that are stained dark blue. (Methanamine silver stain;  100.)

limits. He was treated with intravenous micafungin for 2 weeks followed by oral voriconazole for 6 weeks, to which he responded well. Improvement started within a week of starting intravenous antibiotics. On follow-up visit 6 weeks after he completed oral voriconazole, his range of motion had improved—metacarpophalangeal joint 0/80, proximal interphalangeal joint 0/80, and distal interphalangeal joint 0/50. The swelling and tenderness also improved significantly. This was his final visit with us.

(hyper-IgE immunoglobulinemia).6 Our patient was immunocompetent. After the diagnosis of fungal tenosynovitis was confirmed, he had an immunological work-up.1 All his parameters were normal. The patient had received 5 injections of corticosteroids 2 to 6 weeks apart before presenting to us. Although impossible to directly link his infection to those injections, infection is a well-known risk associated with multiple steroid injections.8 Previously reported Candida tenosynovitis has been from C. albicans, which is a normal commensal in human skin and nails. C. parapsilosis is an emerging pathogen, especially implicated in neonatal intensive care unit candidemia.9 It is the Candida species with the largest increase in incidence since 1990.9 As a normal commensal of human skin, it is often found in the hands of health care workers. In a study of neonatal intensive care unit health care workers, 19% were colonized with C. parapsilosis.10 Any breach in handwashing protocols during the prior procedures or from steroid injections could be responsible for direct inoculation of fungus in the tenosynovium. There is a case report of septic arthritis with C. parapsilosis in which rice bodies were seen at surgical debridement.4 Reviewing the operative report of the second surgery that our patient had at an outside facility, there was mention of a pumpkin seedelike appearance of the nodule that was removed. However, this was not sent for microbiology or culture, and pathology revealed fibrinous debris. Candida is not typically seen in hematoxylin-eosin stains and requires special stains to be seen in tissue sections. Therefore, a tissue culture is the best way to identify it.1

DISCUSSION Although fungus is commonly implicated in superficial hand infections, invasive fungal infection in hand is rare.1 For superficial infections, Candida albicans is the most common offender, although more recently, non-albicans species have been found to be emerging pathogens.2 Deep infections are usually caused by direct inoculation of fungus into the soft tissues of the hand resulting in tenosynovitis, arthritis, or infection of a prosthesis.3,4 Fungal tenosynovitis, although rare, has been reported.5 It is different from acute bacterial tenosynovitis in that it has a slow onset, with swelling and stiffness as prominent clinical features instead of pain.1 Thus it is not uncommon for these patients to have an extensive rheumatologic work-up prior to their diagnosis, as was the case with our patient. Candida tenosynovitis is a rarely reported entity.6,7 The previously described reports of Candida tenosynovitis have been in immunocompromised individuals. One of the patients had acquired immunodeficiency syndrome,7 and another was a patient with Buckley immunodeficiency J Hand Surg Am.

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REFERENCES

It is impossible to pinpoint the exact cause of fungal infection in our patient. He did not have an open wound from his initial injury. He was not immunocompromised, and hence, it is unlikely that the fungal infection was hematogenously disseminated. So, the fungal infection could have been introduced during either of the 2 prior surgeries or at 1 of the multiple injections he received. Our patient underwent extensive work-up for a rheumatological pathology before receiving the correct diagnosis. Once treatment with micafungin followed by voriconazole was instituted, he had remarkable improvement in subjective and objective examination of his left middle finger. Our case report underscores the need for a high index of suspicion for fungal or atypical tenosynovial infections in a patient with insidious presentation who is not responding to conservative management. Hand surgeons should request cultures for fungus and mycobacteria and potassium hydroxide or calcofluor stain in addition to routine bacterial cultures and pathology for such patients.

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1. Patel MR, Malaviya G. Chronic infections. In: Wolfe SWW, Pederson WC, Hotchkiss RN, Kozin SH, eds. Green’s Operative Hand Surgery. 6th ed. Philadelphia, PA: Churchill-Livingstone; 2010:85e140. 2. Fich F, Abarzua-Araya A, Perez M, Nauhm Y, Leon E. Candida parapsilosis and Candida guillermondii: emerging pathogens in nail candidiasis. Indian J Dermatol. 2014;59(1):24e29. 3. Dunkley AB, Leslie IJ. Candida infection of a silicone metacarpophalangeal arthroplasty. J Hand Surg Br. 1997;22(3):423e424. 4. Jeong YM, Cho HY, Lee SW, Hwang YM, Kim YK. Candida septic arthritis with rice body formation: a case report and review of literature. Korean J Radiol. 2013;14(3):465e469. 5. Cucurull E, Sarwar H, Williams CSt, Espinoza LR. Localized tenosynovitis caused by Histoplasma capsulatum: case report and review of the literature. Arthritis Rheum. 2005;53(1):129e132. 6. Yuan RT, Cohen MJ. Candida albicans tenosynovitis of the hand. J Hand Surg Am. 1985;10(5):719e722. 7. Townsend DJ, Singer DI, Doyle JR. Candida tenosynovitis in an AIDS patient: a case report. J Hand Surg Am. 1994;19(2):293e294. 8. Holland C, Jaeger L, Smentkowski U, Weber B, Otto C. Septic and aseptic complications of corticosteroid injections: an assessment of 278 cases reviewed by expert commissions and mediation boards from 2005 to 2009. Dtsch Arztebl Int. 2012;109(24):425e430. 9. Trofa D, Gacser A, Nosanchuk JD. Candida parapsilosis, an emerging fungal pathogen. Clin Microbiol Rev. 2008;21(4):606e625. 10. Saiman L, Ludington E, Dawson JD, et al. Risk factors for Candida species colonization of neonatal intensive care unit patients. Pediatr Infect Dis J. 2001;20(12):1119e1124.

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Vol. 40, May 2015

Candida parapsilosis Tenosynovitis in an Immunocompetent Patient: Case Report and Review of Literature.

We describe a case of fungal tenosynovitis with Candida parapsilosis, which is an uncommonly reported agent causing tenosynovitis. It occurred in an i...
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