06 September 2023: Articles
Rare Pseudotumor in Ceramic-On-Ceramic Total Hip Replacement with Concomitant Fungal Periprosthetic Joint Infection: A Case Report
Challenging differential diagnosis, Rare diseaseMatias Pablo Marcomini1ABCDEFG*, Javaid Iqbal1AEG, Derek Bennett1AEFG
Am J Case Rep 2023; 24:e941164
BACKGROUND: Total hip replacement (THR) is a commonly performed treatment for severe osteoarthritis. In this report, we present the case of a woman who unfortunately suffered 2 severe but rare complications of THRs: a pseudotumor formation on a Delta ceramic-on-ceramic bearing and a fungal periprosthetic joint infection (PJI).
CASE REPORT: In early 2016, a 63-year-old woman underwent an elective left total hip replacement with ceramic-on-ceramic bearing due to severe osteoarthritis. In 2021, she suffered 2 unprovoked DVTs. Therefore, ultrasound (US) Doppler imaging of the left lower limb was performed, which showed a mass close to the iliac vein. After magnetic resonance imaging (MRI) to further examine the mass, a pseudotumor was confirmed. Revision surgery was performed, after which positive swabs for fungal infection were identified, but were not clinically correlated. A few years before, a deep buccal fungal infection was suspected and treated, but never confirmed. The pseudotumor was confirmed by histology samples. A few weeks later, the patient presented again with symptoms of infection, and 2 debridement, antibiotics, and implant retention (DAIR) procedures were performed, in which further positive swabs of Candida parapsilosis were obtained. Currently, the patient is on conservative therapy with long-term antifungal medication since she refused a staged procedure due to personal circumstances.
CONCLUSIONS: In conclusion, this case report documents the first ever reported pseudotumor associated with a ceramic-on-ceramic bearing THR with concomitant fungal PJI. Although it is unlikely for a person to develop 2 rare complications without them being connected, no causal link could be established.
Keywords: Prosthesis-Related Infections, Candidiasis, Granuloma, Plasma Cell, invasive fungal infections, Venous Thrombosis
Total hip replacement (THR) is a commonly performed treatment for severe osteoarthritis, but it is not free of complications, which can lead to early failure of the components [1,2]. In this report we present the case of a woman who unfortunately suffered 2 severe but rare complications of THRs: a periprosthetic fungal infection, which may have promoted the development of an extremely rare pseudotumor on a ceramic-on-ceramic (CoC) THR. Fungal periprosthetic joint infections (PJI) are rare and devastating infections, which have been growing in numbers and are usually related to chronic comorbidities like diabetes or immunosuppression. They also have higher incidence in patients who receive long-term antibiotics in the 3 months before surgery, usually given as an attempt to treat a previous bacterial PJI [1,3]. In the literature, pseudotumors are defined as non-neoplastic and non-infectious masses resulting from a circumscribed fibrous exudate of inflammatory origin, fluid accumulation, or other causes . These are rare complications with a high probability of inducing early failure of the prosthesis. These have been associated in many cases with metal-on-metal (MOM) and sometimes with metal-onpolyethylene total hip replacements (MOP), with only 1 other published case of a pseudotumor in a ceramic-on-ceramic THR [5–7]. The aim of this case report is to raise awareness of pseudotumor and its possible association with fungal PJI. We describe the second ever reported pseudotumor in a CoC THR and propose a hypothesis for further research into whether a fungal infection could react with the CoC bearing surfaces of the THR, increasing the chances of pseudotumor formation.
In early 2016, a 63-year-old woman presented to our orthopedic clinic with severe left hip pain and reduced range of motion, which was interfering with her daily activities. On plain pelvis X-ray, signs of osteoarthritis were clearly shown. At that time, the patient was actively working, fully independent, and non-diabetic, with a past medical history of hypertension, hypercholesterolaemia, Duputryen’s contracture, appendectomy, oophorectomy, haemorrhoidectomy, and right THR. She was a non-smoker and an occasional drinker. Therefore, she underwent an elective left total hip replacement, for which we implanted a Trilock femoral component size 6, a Biolox Delta ceramic head size 36+8.5 mm, a Pinnacle acetabular component size 52 mm inner diameter, and a Biolox Delta ceramic liner size 52 mm outer diameter and 36 mm inner diameter. All the components were manufactured by DePuy Synthes, Raynham, MA, USA (Figure 1). The procedure was performed without any acute complications, and her postoperative rehabilitation was uneventful.
In May 2018, she presented to her general practitioner (GP) with a suspicious widespread, foul-smelling, white lesion on her tongue, which did not bleed when scratched. Her GP diagnosed her with a fungal infection and started oral antifungal therapy and referred her to the Ear, Nose, and Throat) (ENT) Department. On attendance, the lesion did not resolve after 1 course of fluconazole and a second course of Mycostatin. Therefore, the ENT surgeon, who believed it was a deep-site infection on the floor of the mouth, arranged a biopsy. The biopsy, taken from the anterior and middle part of the tongue, showed squamous cell epithelium with hyperkeratosis, acanthosis, and subepithelial mild chronic inflammation, without signs of fungal infection on periodic acid Schiff (PAS) stain. These results were as expected since she had already received 2 cycles of antifungal medication, and we wonder if this event could be the related to the later PJI.
During 2019, our patient was admitted due to thigh-to-calf leg swelling, with severe tenderness on the deep calf area and cramping sensation. Deep vein thrombosis was suspected; therefore, an US Doppler scan of the deep venous system was performed, which showed a 12.7×5-cm mass in the iliac region lateral to the left hip, which was thought to be a hematoma (Figure 2). She also had non-patent, non-compressible superficial and common femoral veins, suggesting deep vein thrombosis (DVT). The popliteal vein was patent and compressible, but the external iliac vein was not visualized. Following this finding, a CT scan of the thorax, abdomen, and pelvis (CT-TAP) with contrast was performed due to concerns of malignancy since the patient had an unprovoked DVT and a mass in the hip area. The CT-TAP showed no signs of malignancy and confirmed that the DVT had its origin in the external iliac vein. She was treated only with oral anticoagulant medication, since no hematological pathology was detected. Few months later, the patient was still suffering from left thigh-to-calf swelling, regardless of the ongoing anticoagulation and compression stockings. Following this, a CT venogram of the leg was performed (Figure 3), which showed another collection, this time 10×6×7cm, and no signs of DVT. For further screening, an ultrasound and magnetic resonance venogram was performed, to further evaluate the mass next to the THR. At the same time, to treat the long standing DVT, an external iliac vein stent was inserted (Figure 4, arrow 1).
Soon thereafter, an ultrasound was performed due to suspicion of a pseudotumor. A second MRI with MARS views was performed by request of the orthopedic consultant, confirming a pseudotumor in November 2020.
With this information, our orthopedic consultant performed a revision surgery of her left THR on 24 February 2022, which revealed a large pseudotumor. The bearing was exchanged for a ceramic-on-polyethylene bearing because the small ceramic debris that this prothesis created could have created the inflammation necessary to form a pseudotumor. While the friction of 2 hard surfaces like ceramic-on-ceramic can cause debris, a hard surface like ceramic will not crumble against a soft surface like polyethylene . The pseudotumor was debulked but not completely excised due to the risk of iatrogenic injury to adjacent neurovascular structures (Figure 4, arrow 2). Specimens were sent for microbiology culture, which was positive for
Fungal PJI are rare and can be difficult to diagnose. They account for approximately 1% of the total number of PJIs . In addition to this, they are usually slow-developing infections, taking on average 86 months to present symptoms according to some minor studies and systematic reviews, and blood inflammatory markers are rarely elevated . In our case, the fungal PJI did not show any symptoms before the joint was revised, with the pseudotumor, leg swelling, and pain being the main indications for the revision 6 years after the primary procedure. Moreover, even after obtaining a revision sample with a growth of
Looking at the main premise of our case report, we have would like to introduce the hypothesis that our novel, second case ever described, finding of a pseudotumor in a CoC THR, was promoted by an interaction of the fungal species with the ceramic components of the liner. The exact pathophysiology of pseudotumor formation in this case is still uncertain. Pseudotumors have a histopathological presentation similar to that of an aseptic lymphocytic vasculitisassociated lesion, which can be related to metal hypersensitivity, unlike in our case, or a reaction to excess wear debris, which we can presume is our case, since there is no metal present in the contact surfaces of the CoC THR . According to the FDA, free-metal ions might play an important role in this . Pseudotumors are common in metal-on-metal (MOM) hip replacements, having an incidence rate of around 32%, depending on the study. There is also a wide array of reported pseudotumors in metal-on-polyethylene (MoP) and ceramic-on-polyethylene (CoP), but there is only 1 other case of pseudotumor in a ceramic-on-ceramic (CoC) THR . The main question regarding CoCs is how a pseudotumor could be created in a prosthesis with low capacity to shed metal. We believe that ceramic could also create this local reaction, as reported previously .
Although the statistical likelihood of having 1 rare complication and 1 extremely rare complication of total hip replacements in the same patient suggests an association between them, we have not found any research supporting this.
Finally, this case report also describes an interesting symptom of pseudotumor formation, which is related to the mass effect, compressing other tissues. In this case, compressing the femoral vein produced a large unprovoked DVT in an otherwise healthy patient, which occurs by interfering with the principles of the Virchow triad, as reported previously .
This case report documents for first time the presence of a pseudotumor with a concomitant fungal PJI on CoC THR. Although it is unlikely for a person to develop 2 rare complications together without them being connected, we were unable to draw any definitive association due to the uniqueness of the case. Therefore, we would like to encourage more research on how Candida species react with ceramic surfaces or wait for more case like this to be reported. In this case report we also tried to raise awareness about the etiology, pathophysiology and symptoms of pseudotumors, as well as the behavior of fungal infections in prostheses, which although rare, are growing in numbers.
FiguresFigure 1.. X-ray of day 1 after primary total hip replacement with a ceramic-on-ceramic bearing. Figure 2.. Ultrasound Doppler performed for suspicion of deep venous thrombosis, showing a large pseudotumor mass. Figure 3.. CT venogram showing pseudotumor. Figure 4.. X-Ray day 1 after revision of left total hip replacement, ceramic-on-polyethylene bearing [arrow 2]. Vena cava stent in situ [arrow 1]. Figure 5.. H&E section 10×. Area of hyaline fibrous tissue. Pseudotumor. Figure 6.. H&E section 10×. Edematous area with perivascular lymphocytic infiltration. pseudotumor. Figure 7.. H&E section 10×. Diffuse and perivascular inflammation [arrow 1] with scattered multinuclear giant cells [arrow 2].
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