Mucormycosis May Mimic Disease Relapse in Wegener’s Granulomatosis

Nogueira, Estela; Ind, Philip W.; Friedland, Jon S.; Salama, Alan D.

doi:10.3899/jrheum.091423

Cited by 14 publications

(11 citation statements)

References 3 publications

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“…There are few cases of systemic lupus erythematosus associated mucormycosis in the literature, but the mortality of these infections is exceedingly high (80%) [50]. There have also been reports of mucormycosis in other autoimmune diseases, where the infection mimicked the relapse of Wegener's granulomatosis [51] or, in another case, was confused with giant cell arteritis [52]. Although these cases are extremely rare, they may be underdiagnosed and mucormycosis should be included in the differential diagnosis.…”

Section: Corticosteroids and Other Immunosuppressive Agentsmentioning

confidence: 99%

Epidemiology and Diagnosis of Mucormycosis: An Update

Skiada

Pavleas

Drogari‐Apiranthitou

2020

JoF

450

508

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Mucormycosis is an angioinvasive fungal infection, due to fungi of the order Mucorales. Its incidence cannot be measured exactly, since there are few population-based studies, but multiple studies have shown that it is increasing. The prevalence of mucormycosis in India is about 80 times the prevalence in developed countries, being approximately 0.14 cases per 1000 population. Diabetes mellitus is the main underlying disease globally, especially in low and middle-income countries. In developed countries the most common underlying diseases are hematological malignancies and transplantation. Τhe epidemiology of mucormycosis is evolving as new immunomodulating agents are used in the treatment of cancer and autoimmune diseases, and as the modern diagnostic tools lead to the identification of previously uncommon genera/species such as Apophysomyces or Saksenaea complex. In addition, new risk factors are reported from Asia, including post-pulmonary tuberculosis and chronic kidney disease. New emerging species include Rhizopus homothallicus, Thamnostylum lucknowense, Mucor irregularis and Saksenaea erythrospora. Diagnosis of mucormycosis remains challenging. Clinical approach to diagnosis has a low sensitivity and specificity, it helps however in raising suspicion and prompting the initiation of laboratory testing. Histopathology, direct examination and culture remain essential tools, although the molecular methods are improving. The internal transcribed spacer (ITS) region is the most widely sequenced DNA region for fungi and it is recommended as a first-line method for species identification of Mucorales. New molecular platforms are being investigated and new fungal genetic targets are being explored. Molecular-based methods have gained acceptance for confirmation of the infection when applied on tissues. Methods on the detection of Mucorales DNA in blood have shown promising results for earlier and rapid diagnosis and could be used as screening tests in high-risk patients, but have to be validated in clinical studies. More, much needed, rapid methods that do not require invasive procedures, such as serology-based point-of-care, or metabolomics-based breath tests, are being developed and hopefully will be evaluated in the near future.

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Section: Corticosteroids and Other Immunosuppressive Agentsmentioning

confidence: 99%

Epidemiology and Diagnosis of Mucormycosis: An Update

Skiada

Pavleas

Drogari‐Apiranthitou

2020

JoF

450

508

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“…This case also illustrates that symptoms caused by invasive fungal infections can mirror disease progression or relapse in GPA [22,23]. The patient presented with hemoptysis and dyspnea suggestive of relapse of GPA; however, pulmonary mucormycosis or other infections may also have a similar presentation.…”

Section: Discussionmentioning

confidence: 76%

A Fatal Case of Treatment-Related Adverse Effects in Granulomatosis with Polyangiitis (Wegener’s Granulomatosis)

Marina¹,

Maria²

2011

OJRA

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Well-studied therapies have proven to be effective in treating Granulomatosis with polyangiitis (formerly Wegener's granulomatosis) (GPA). There has been considerable improvement in survival of patients with GPA but treatment related morbidity and mortality remains still high, particularly in patients with renal disease. We describe a case of 64-year old woman with recent onset GPA, who responds well to the initial cyclophosphamide based therapy but latter develops a fatal stroke. Infectious complications should be considered in patients with GPA who are on adequate immunosuppression but develop symptoms that may mimic a relapse. Aggressive diagnostic interventions should be undertaken to discriminate between an infection and a relapse of GPA.On autopsy, right lung revealed diffuse necrosis and inflammation. There was cavitation of the lower lobe associated with thrombotic occlusion of the pulmonary artery and vein branches supplying the area. Non-septated, irregularly wide (6 m to 50 m) fungal hyphae

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“…Berenguer et al . [15] and Noguiera[16] et al . have also reported pulmonary mucormycosis mimicking disease relapse in patients with WG on immunosuppressive therapy.…”

Section: Discussionmentioning

confidence: 99%

Granulomatosis with polyangitis with mononeuritis multiplex-immunosuppressives playing a double-edged sword

et al. 2014

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A 52-year-old female was diagnosed with rheumatoid arthritis and was on methotrexate and prednisolone. She developed fever, cough, hemoptysis, and cavitary lesion on chest skiagram. She was put on antitubercular therapy without any improvement, meanwhile she developed painful right foot drop. Clinicoradiology and C-ANCA study confirmed the diagnosis of granulomatosis with polyangitis (GPA). She was started on cyclophosphamide, corticosteroid, and co-trimoxazole. While her treatment was being continued she showed significant improvement of pulmonary manifestations. About 1 year later, there was reappearance of fever, cough, and radiological opacity with oropharyngeal candidiasis. She became very ill with disseminated intravascular coagulation (DIC)-like features. Immunological markers were negative but bronchoalveolar lavage fluid study showed growth of Aspergillus spp. The patient was promptly put on intravenous voriconazole but unfortunately she succumbed to her illness.

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Mucormycosis May Mimic Disease Relapse in Wegener’s Granulomatosis

Cited by 14 publications

References 3 publications

Epidemiology and Diagnosis of Mucormycosis: An Update

Epidemiology and Diagnosis of Mucormycosis: An Update

A Fatal Case of Treatment-Related Adverse Effects in Granulomatosis with Polyangiitis (Wegener’s Granulomatosis)

Granulomatosis with polyangitis with mononeuritis multiplex-immunosuppressives playing a double-edged sword

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