Xanthogranulomatous osteomyelitis of pubic bone mimicking neoplasm: a case report and literature review.

BACKGROUND: Xanthogranulomatous osteomyelitis (XO) is a rare disease characterized radiologically by an osteolytic lesion with cortical expansion or disruption. Differentiating this condition from other osteolytic diseases such as primary or metastatic bone neoplasms is imperative. Several case reports have been published on XO, with previous reports predominantly identifying bacteria such as Pseudomonas or Staphylococcus as causative organisms. However, fungal infection-induced XO has not yet been reported. CASE PRESENTATION: We present the case of a 23-year-old woman with a tumor-like osteolytic lesion in the pubic bone. The patient had experienced pelvic pain and intermittent febrile episodes for 2 months. Plain radiography revealed an osteolytic lesion in the right pubic tubercle. Magnetic resonance imaging suggested a cystic bone tumor or tubercular infection. Surgical intervention included curettage of the lesion and irrigation with normal saline. Histopathological examination of the specimen revealed abundant foamy histiocytes with inflammatory infiltrates consistent with XO. Culture of the osteolytic lesion confirmed an Aspergillus species infection and antifungal treatment was initiated. At 1-year follow-up, no evidence of local recurrence was observed. CONCLUSIONS: Although rare, XO requires differentiation from similar conditions and is treated with surgical intervention and targeted medical therapy based on the identified organisms. Clinicians should be mindful that XO can also be induced by fungal infections and that combination antifungal treatments may be beneficial in such cases.

Number of sinus procedures reduces inpatient mortality for invasive fungal rhinosinusitis.

OBJECTIVES: To evaluate surgical outcomes of invasive fungal rhinosinusitis MATERIALS AND METHODS: The National Inpatient Sample Database (2000-2015 Q3) was queried for patients with a diagnosis of aspergillosis and/or mucormycosis and a diagnosis of acute sinusitis using the International Classification of Diseases, Ninth Edition. Factors associated with inpatient mortality were then identified with multivariate logistic regression. RESULTS: 514 adult patients with a median age of 57.0 years were identified, of which 231 (44.9 %) underwent sinus surgery. Surgical patients had a longer length of stay (17.0 vs 9.0 days, p < 0.001) and higher total charges ($139,762.00 vs $57,945.00, p < 0.001). The number of sinus procedures was associated with reduced odds of inpatient mortality (OR 0.69; p < 0.001) in multivariate analysis. Hypertension (OR 0.34, p = 0.002) and chronic kidney disease (OR 0.23, p = 0.034) were associated with reduced odds of inpatient mortality. Total number of procedures (OR 1.24; p = 0.002), mucormycosis (OR 2.75, p = 0.002), age (OR 1.03, p = 0.006) and acid-base disorders (OR 2.85, p = 0.012) were associated with increased odds of inpatient mortality. CONCLUSION: This represents the first large scale study to evaluate outcomes for invasive fungal rhinosinusitis. These findings suggest the odds of inpatient mortality decrease with greater extent of sinus surgery performed. The potentially protective roles of hypertension and chronic kidney disease should be evaluated in future research.

Appropriate Total Rehabilitation in the Bilateral Maxillectomy Defected Diabetic Patient Secondary to Aggressive Aspergillosis.

Aggressive aspergillosis is a life-threatening fungal infection with rapid progress, mainly affecting the maxillofacial area, especially the nose and paranasal sinuses, in patients with immunocompromised conditions such as diabetes mellitus. Aggressive aspergillosis infection should be differentiated from other invasive fungal sinusitis for early recognition with correct prompt treatment. The main treatment is aggressive surgical debridement such as maxillectomy. Although aggressive debridement should be performed, preservation of the palatal flap should be considered for better postoperative outcomes. In this manuscript, we report the case of a diabetic patient with aggressive aspergillosis affecting the maxilla and paranasal sinuses and describe the appropriate surgical management and prosthodontic rehabilitation.

Orbital Apex Syndrome Caused by Intraorbital Aspergillus Infection.

Orbital apex syndrome, a clinical disease that is uncommon and has a high fatality rate. Tumor, endocrine, and inflammatory variables are frequently responsible for its occurrence. The authors describe a 53-year-old Chinese man who was diagnosed with orbital apex syndrome and coupled type 2 diabetes mellitus and a fungus infestation. Treatment included nasal endoscopic orbital apical decompression, anti-infection, and adequate debridement. Except for inevitable optic nerve damage, postoperative proptosis and headache manifestations improved, and systemic infection was timely contained with no signs of recurrence or serious complications occurred. The orbital apex syndrome is difficult to treat, and soon as possible biopsy of the lesion, aggressive surgical decompression, and antifungal treatment seem to be effective ways to improve survival rates.

[Intralobar Sequestration Associated with Aneurysm of the Aberrant Artery and Aspergillus Infection: Report of a Case].

A 32-year-old man was referred to our department for surgery because of recurrent pneumonia. Aspergillus fumigatus was detected in his sputum culture at the first visit. We started antifungal therapy one month before surgery. His chest radiograph showed an infiltrative shadow in the right lower lobe, and chest computed tomography (CT) showed an infiltrative shadow and large and small cystic changes in the right lower lobe. CT angiography (CTA) revealed two anomalous arteries supplied by the aorta that drained into the right lower lobe. An aneurysm with a diameter of 25 mm had formed in one anomalous artery. Based on these findings, intralobar pulmonary sequestration with Aspergillus infection and an anomalous artery forming an aneurysm was diagnosed. In addition, we embolized the aneurysm of the anomalous artery. After embolization, right lower lobectomy was safely performed. The patient was discharged on the 15th post-operative day with no complications.

[Cavernostomy for Extensive Intrapulmonary Cavitary Abscesses That Turned into Destroyed Lungs].

Surgical resection of the infected lung with curative intent is the treatment of choice for lung abscesses that are difficult to control with medical treatment alone. However, lung resection is considered difficult in some cases. Herein, we report two cases of destroyed lungs with severe symptoms, for which palliative cavernostomy was performed instead of infected lung resection. Case 1 was a 45-year-old man who had granulomatosis with polyangiitis in both lungs. Steroid pulse and immunosuppression therapies were repeated, resulting in a huge, destroyed lung on the right side with chronic necrotizing bilateral aspergillosis, causing severe symptoms. Considering the bilateral spread and extension of the cavity lesions, cavernostomy was performed for the destroyed right lung. Case 2 was a 73-year-old woman who had undergone a left lower lobectomy for a metastatic lung tumor and developed a destroyed lung with severe symptoms in the residual left upper lobe caused by a non-tuberculous mycobacterial infection. Since a completion pneumonectomy with curative intent was considered too invasive for her poor general condition, cavernostomy was performed for the destroyed lung. Palliative operations significantly relieved the severe symptoms and improved the general conditions of these patients, enabling outpatient follow up.

Aspergillus spp. osteoarticular infections: an updated systematic review on the diagnosis, treatment and outcomes of 186 confirmed cases.

Aspergillus spp. osteoarticular infections are destructive opportunistic infections, while there is no clear consensus on their management. The purpose of this review is to investigate the current literature regarding Aspergillus spp. osteoarticular infections. An electronic search of the PubMed and Scopus databases was conducted considering studies that assessed osteoarticular infections from Aspergillus spp. We included only studies with biopsy proven documentation of positive cultures or histological findings for Aspergillus spp., and those with essential information for each case such as the anatomical location of the infection, the type of treatment (conservative, surgical, combination), the antifungal therapy, and the outcome. Overall, 148 studies from 1965 to 2021 including 186 patients were included in the review. One hundred and seven (57.5%) patients underwent surgical debridement in addition to antifungal therapy, while 79 (42.7%) patients were treated only conservatively. Complete infection resolution was reported in 107 (57.5%) patients, while partial resolution in 29 (15.5%) patients. Surgical debridement resulted in higher complete infection resolution rate compared to only antifungal therapy (70.0% vs. 40.5%, P < 0.001), while complete resolution rate was similar for antifungal monotherapy and combination/sequential therapy (58.3% vs. 54.5%; P = 0.76). Last, complete resolution rate was also similar for monotherapy with amphotericin B (58.1%) and voriconazole (58.6%; P = 0.95). The results of this study indicate that antifungal monotherapy has similar efficacy with combination/sequential therapy, while voriconazole has similar efficacy with amphotericin B. Moreover, surgical debridement of the infected focus results in better outcomes in terms of infection eradication compared to conservative treatment. LAY SUMMARY: Antifungal monotherapy has similar efficacy with combination/sequential therapy, and voriconazole has similar efficacy with amphotericin B for the treatment of Aspergillus spp. osteoarticular infections, while surgical debridement of the infected focus improves the infection eradication rate.

Aspergillosis of the Maxillary Sinus Associated With Dental Implant.

Aspergillosis is a fungal disease caused by the fungus Aspergillus; this disease frequently involves the lungs and occasionally the maxillary sinus. Aspergillosis in the maxillary sinus usually has the characteristics of a noninvasive form. It has been suggested that spores of aspergillus can be inhaled into the maxillary sinus via the osteomeatal complex or via an oroantral fistula after dental procedures, such as an extraction. However, maxillary aspergillosis related to implant installation has rarely been reported. This report regards unusual cases of maxillary aspergillosis associated with dental implant therapies in healthy patients. The cases were successfully treated with the surgical removal of the infected or necrotic tissues.

Surgical resection for persistent localized pulmonary fungal infection prior to allogeneic hematopoietic stem cell transplantation: Analysis of six cases.

OBJECTIVE: Although invasive fungal disease (IFD) is an important complication in allogeneic hematopoietic stem cell transplantation (HSCT), the clinical significance of surgery, including the role of surgical resection for persistent pulmonary fungal disease prior to allogeneic HSCT in the current era with a variety of available antifungal agents, is controversial. We investigated the role of surgical resection. METHODS: We retrospectively investigated six patients who underwent surgical resection of suspected pulmonary fungal disease prior to allogeneic HSCT between April 2007 and June 2016 at our medical center. RESULTS: We present six patients who underwent surgical resection of suspected pulmonary fungal disease prior to allogeneic HSCT. In our case series, three of four patients who were given a presurgical diagnosis of possible IFD were given a proven diagnosis after surgery, including two cases of invasive aspergillosis (IA) and one case of mucormycosis. All surgeries were performed by video-assisted thoracic surgery (VATS) for lobectomy without major complications. Recurrence of IFD was not observed after allogeneic HSCT in any of the six patients. CONCLUSION: Our experience indicated that surgical resection of persistent localized pulmonary lesions of IFD before allogeneic HSCT was helpful for obtaining a definitive diagnosis and might be useful for reducing recurrence after HSCT.

Aspergillus flavus native valve endocarditis following combined liver and renal transplantation: Case report and review of the literature.

Aspergillus endocarditis is a rare infection that occurs most commonly in patients with prior cardiac surgery but cases in post-transplant recipients without prior cardiac surgery have been reported. Diagnosis is often delayed and requires high index of suspicion. We here report a case of Aspergillus endocarditis in solid organ transplant recipient.

[Contiguous cutaneous inflammation secondary to an aspergillus sinusitis]. / Inflammation cutanée de contiguïté secondaire à une sinusite aspergillaire.

BACKGROUND: Contiguous skin inflammation is a poorly described entity. It constitutes a cutaneous manifestation of an underlying ongoing process (infectious, inflammatory or neoplastic). Sinusitis is a known cause. PATIENTS AND METHODS: We report the case of a 70-year-old patient consulting for an ongoing centrofacial inflammatory plaque. Cutaneous biopsy revealed a polymorphic inflammatory infiltrate, and cutaneous microbiological specimens were negative. A facial CT-scan showed left maxillary sinusitis. Intra-sinus samples obtained at surgery showed aspergillus. Voriconazole combined with maxillary sinus surgery resulted in healing of the facial plaque. DISCUSSION: There have been only two published cases of contiguous skin inflammation related to sinusitis but no reported cases caused by aspergillus sinusitis. Herein we report the third case of contiguous skin inflammation associated with sinusitis, which is also the first related to aspergillus sinusitis.

Fungal Endocarditis Due to Aspergillus oryzae: The First Case Reported in the Literature.

Infective endocarditis (IE) is a severe disease with high mortality and morbidity. Prosthetic valve endocarditis is a life-threatening complication which can occur in less than 10% of patients with valve prosthesis. A fungal etiology of IE is rare and accounts for only 2-4% of all case of endocarditis, but is associated with a higher mortality and morbidity. Herein is reported the first case of fungal endocarditis of aortic valve prosthesis due to Aspergillus oryzae in a 67-year-old caucasian man who nine years previously underwent mitral and aortic valve replacement with mechanical prostheses, and tricuspid annuloplasty for acute IE due to Enterococcus spp. Seven months previously, the patient also underwent a redo cardiac procedure to replace a mitral valve prosthesis with a new mechanical device due to a leakage. Aspergillus oryzae showed impressive growth with strong and unexpected virulence in both local and systemic settings.

Successful Antifungal Combination Therapy and Surgical Approach for Aspergillus fumigatus Suppurative Thyroiditis Associated with Thyrotoxicosis and Review of Published Reports.

In immunocompromised patients, Aspergillus infections are important causes of morbidity and mortality. We describe a patient with cryoglobulinemic vasculitis who developed disseminated invasive aspergillosis with thyrotoxicosis caused by Aspergillus fumigatus. The diagnosis was based upon radiological, microbiological and pathological findings. The patient was treated successfully with voriconazole and caspofungin treatment followed by total thyroidectomy. We provide an overview of published reports on Aspergillus thyroiditis with an emphasis on therapeutic approaches.

Core curriculum illustration: invasive fungal sinusitis.

This is the 27th installment of a series that will highlight one case per publication issue from the bank of cases available online as part of the American Society of Emergency Radiology (ASER) educational resources. Our goal is to generate more interest in and use of our online materials. To view more cases online, please visit the ASER Core Curriculum and Recommendations for Study online at http://www.erad.org/?page=CCIP_TOC .

Advanced surgical technique for invasive fungal sinusitis: endoscopic orbit-sinus combined approach.

BACKGROUND: Invasive fungal sinusitis is usually associated with poor prognosis, but no clear guidelines have been established for surgical treatment. Here, we report the development and application of the endoscopic orbit-sinus combined approach (EOSCA), a novel surgical technique to approach the nasal cavity and orbit concurrently, in patients with invasive fungal sinusitis with orbital infiltration. MATERIAL AND METHODS: Two patients with invasive fungal sinusitis infiltrating the orbit underwent EOSCA. Transnasal endoscopy was performed for maximum debulking of tissues infiltrated by fungi in the nasal cavity and orbit, before making an incision into the palpebral conjunctiva. An endoscope was then inserted into the orbit through the incision in the palpebral conjunctiva to remove adipose tissue and muscles that had been infiltrated by fungi from the orbital regions where the transnasal approach was difficult or impossible. Another surgeon assisted the procedure by operating an endoscope concurrently via the nasal cavity (four-hands technique). RESULTS: We were able to remove lesions safely and with precision, preserving visual acuity and a functional eyeball in both cases. Currently, the patients are alive, with no postoperative complications, recurrence, or disfigurement. CONCLUSIONS: This novel method shows promise as a safe and reliable surgical procedure for patients with invasive fungal sinusitis infiltrating into the orbit, with no postoperative complications, recurrence, or disfigurement.

Management of granulomatous cerebral aspergillosis in immunocompetent adult patients: a review.

Cerebral aspergillosis, is an infrequent, opportunistic infection of the central nervous system that accounts for 5-10% of all intracranial fungal pathology. It is uncommon in immunocompetent patients and has a significant disease burden, with high morbidity and mortality, even with appropriate treatment. Basic principles of abscess management should be employed, including aspiration and targeted anti-fungal therapy for 12-18 months. However, reported outcomes with a purely minimally invasive approach are poor and there should be a low threshold for surgical excision, especially in resource poor settings and in patients with deteriorating neurology harbouring sizeable masses. Evidence favouring gross total excision over subtotal resection is lacking, however. It is notable that these recommendations are largely based on retrospective case series and isolated case reports. There is a need therefore for international collaboration to evaluate management strategies for immunocompetent patients with cerebral aspergillosis.

Aspergillus Osteomyelitis of the Skull.

Osteomyelitis of the craniofacial skeleton is rare, with fungal pathogens least commonly implicated. The authors present 2 patients of osteomyelitis of the skull caused by Aspergillus spp. and discuss the diagnosis, clinicopathological course, and management strategies.Late recurrence seen in this type of infection warrants long-term follow-up and a high index of suspicion for the clinical signs associated with recurrence.Such patients would benefit from their surgical debridement being planned and managed via a specialist craniofacial unit, so as to utilize the most aesthetically sensitive approach and the experience of specialists from several surgical disciplines.

Sphenoid sinus aspergilloma in trans-sphenoidal surgery for pituitary adenomas.

BACKGROUND: Simultaneous appearance of sphenoid sinus aspergilloma and pituitary adenoma is a very rare finding. METHODS: Retrospective analysis of patients with sellar and sphenoid sinus mass lesions who underwent trans-sphenoidal surgery was performed. Demographic data, medical history, predisposing factors, clinical picture, neurological status and radiological findings were reviewed. All patients underwent a trans-sphenoidal microsurgical treatment, and acquired specimens underwent both histopathological and microbiological analysis. RESULTS: Sphenoid sinus aspergilloma was encountered in seven patients. Three patients had an isolated sphenoid sinus aspergilloma and four patients with pituitary macroadenoma had a sphenoid aspergilloma as an incidental finding. CONCLUSIONS: Sphenoid sinus aspergilloma can be found during trans-sphenoidal surgery for pituitary adenomas. Sphenoid sinus extirpation followed by adenomectomy is the treatment of choice unless invasive aspergilloma is encountered requiring additional antifungal therapy.

The Role of Adjunctive Therapies in the Management of Invasive Sino-Orbital Infection.

OBJECTIVE: Invasive sino-orbital fungal infections are life-threatening complications of immunonosupression that are difficult to treat. Currently there are no standard treatment guidelines. The most widely accepted therapy includes parenteral anti-fungal therapy and surgical debridement of sinuses with orbital exenteration, a procedure that is not only disfiguring, but may increase morbidity. Injection of retrobulbar Liposomal Amphotericin B (L-AMB) is an alternative approach that provides local administration to infected tissues. The adjunct use of anti-fungal retrobulbar injections not been extensively reviewed in treating sino-orbital infection. We are reporting the multimodal approach of using L-AMB retrobulbar injections in combination with sinus debridement, intravenous (IV) anti-fungal therapy, and hyperbaric oxygen (HBO) for the management of sino-orbital infection. METHOD & RESULTS: Review of literature of 12 cases and retrospective evaluation of one patient with sino-orbital Aspergillus flavus infection on chemotherapy for T-cell acute lymphocytic leukemia treated with retrobulbar Amphotericin B, IV anti-fungal agents, and hyperbaric oxygen therapy. Clinical characteristics, radiographic features, management techniques, and clinical outcomes are described. CONCLUSION: Retrobulbar Amphotericin B injection may be an effective adjunct to hyperbaric oxygen and parenteral anti-fungals in the control of sino-orbital fungal infections.