Tracheal, laryngeal and pulmonary mucormycosis followed by organizing pneumonia in a patient with Adult Onset Still's Disease

Abstract

We report a case of tracheal, laryngeal and pulmonary mucormycosis in a patient receiving immunosuppressive medication for an autoinflammatory fever syndrome. Mucormycosis was confirmed by histopathology from tracheal specimens and molecular evidence of Lichtheimia.A surgical approach was not possible because of the multifocal disease pattern and the extent of tracheal involvement. The patient was successfully treated with liposomal amphotericin B followed by posaconazole maintenance therapy. After 9 months, recurrent pulmonary mucormycosis was suspected but emerged as organizing pneumonia without evidence of active fungal infection.

The relationship between organising pneumonia and invasive mould disease in patients with haematologic malignancy

“…In addition, the secondary OP-associated infections may be attributed to higher mortality. Since the combination therapy of antimicrobial agents and steroids is recommended for the management of secondary OPassociated infections, [13][14][15][16] disease control may not be achieved with only one of these. Although fungal hyphae were not found, most patients (12/15, 80%) confirmed as OP also received antifungal therapy in this study.…”

“…[9][10][11][12] However, OP associated with invasive mould disease has only been sporadically reported. Concomitant occurrence of invasive pulmonary aspergillosis (IPA), 13 chronic pulmonary aspergillosis leading to the development of OP, 14 pulmonary mucormycosis (PM) followed by OP, 15 and invasive airway aspergillosis with OP 16 were described as case reports. These cases indicate that invasive mould disease may be involved with the pathogenesis of OP.…”

Isolated tracheobronchial mucormycosis: Report of a case and systematic review of literature

“…Cases, where lung collapse was observed on chest radiograph along with an exophytic tracheal or central airway lesion, were also included if the radiograph normalised immediately after managing tracheal mucormycosis. We excluded cases with concomitant pulmonary parenchymal involvement or tracheal infiltration secondary to mucormycosis of the adjacent structures (such as laryngeal, pharyngeal, oesophageal, mediastinal structures or vascular aneurysmal rupture into the trachea) 4–9 . We also excluded cases with lobar or segmental bronchial involvement 10–12 .…”

“…We excluded cases with concomitant pulmonary parenchymal involvement or tracheal infiltration secondary to mucormycosis of the adjacent structures (such as laryngeal, pharyngeal, oesophageal, mediastinal structures or vascular aneurysmal rupture into the trachea). [4][5][6][7][8][9] We also excluded cases with lobar or segmental bronchial involvement. [10][11][12] We excluded conference abstracts, review articles and editorials.…”

Coronavirus Disease 2019–Associated Mucormycosis: Risk Factors and Mechanisms of Disease

“…In addition, the hyperferritinemic state can sustain a feed forward loop of hyperinflammation and also modulate the lymphocyte response (26). Indeed, there have been case reports of mucormycosis in hyperferritinemic syndromes like hemophagocytic lymphohistiocytosis (27) and adult onset Still's disease (28).The prothrombotic state…”