Rare complication of bronchoesophageal fistula due to pulmonary mucormycosis after induction chemotherapy for acute myeloid leukemia: a case report

Abstract

BackgroundMucormycosis is a rare and life-threatening invasive fungal infection. Pulmonary mucormycosis commonly occurs in patients with severe neutropenia. Typically, pulmonary mucormycosis causes tissue necrosis resulting from angioinvasion and subsequent thrombosis, so most cases can occur with necrotizing pneumonia and/or hemoptysis. Some complex cases may invade adjacent organs, such as the mediastinum, pericardium, and chest wall. However, to the best our knowledge there is little known regarding bronchoesophageal fistula due to pulmonary mucormycosis after induction chemotherapy for acute myeloid leukemia. We present a case report about this unusual presentation.Case presentationA 51-year-old Korean man was diagnosed as having acute myeloid leukemia and received induction chemotherapy. After prolonged severe neutropenia, he complained of coughing with aspiration. Imaging showed a bronchoesophageal fistula with extensive necrotizing pneumonia in the middle and lower lobes of his right lung. Bronchoscopy showed near total tissue necrosis in the middle lobe of his right lung, creating an orifice. A bronchial scope was passed through and was able to be connected with his esophagus; a bronchial wall biopsy was performed. Esophagoscopy revealed a large linear defect of his esophageal wall 30 cm from the incision that may have connected with the bronchus. A bronchial biopsy showed typical hyphae with necrotic tissue, indicating pulmonary mucormycosis. He was given amphotericin B, and a wide excision of lung and esophagus was planned. However, he suddenly died due to massive hemoptysis.ConclusionHere we present an extremely rare case of bronchoesophageal fistula with severe necrotizing pneumonia due to pulmonary mucormycosis.

Fungal Infections Complicating Lung Transplantation

“…[24][25][26] IA is almost exclusively a disease of immune suppressed or immune deficient patients. Specific risk factors include hematological malignancies with neutropenia; [27][28][29] HSCT (especially with GVHD); 14,22,23,[30][31][32][33][34] SOT; 8,23,31,[34][35][36][37][38][39][40][41][42][43][44] immunosuppression; [45][46][47] iron overload states; [48][49][50][51][52][53] and cytomegalovirus disease. 49,54 In one prospective study of the cumulative incidence of IA in organ transplant recipients (OTRs), the most commonly affected patients were recipients of allogeneic HSCT (2.9%/year), followed by lung transplant (2.4%/year), heart transplant (0.8%/ year), autologous HSCT (0.5%/year), liver transplant (0.3%/year), and renal transplant (0.1%/year).…”

“…Risk factors for mucormycosis include poorly controlled diabetes; 39,112,[120][121][122][123][124] hematological malignancies with neutropenia; 123,[125][126][127][128][129][130] HSCT; 14,22,[32][33][34] SOT; 8,34,[38][39][40][41][42][43][44] immunosuppression; 131,132 human immunodeficiency virus (HIV) infection; 111,133-136 peritoneal dialysis; [137][138][139] iron overload states; [51][52][53] and prior receipt of VCZ. 33,123,[140][141][142] Mucormycosis rarely affects immunocompetent persons, but cutaneous, rhino-orbital, and (rarely) disseminated infections may occur following local cutaneous or soft tissue trauma.…”

“…115 Dissemination is usually fatal. 107,112,115 Based on anatomic localization, mucormycosis can be classified as one of six forms: 110 (1) rhino-orbital-cerebral, 39,124,147 (2) pulmonary, 38,125,136 (3) cutaneous, 170,179,180 (4) gastrointestinal, 181,182 (5) disseminated, 32,38,107,112 and (6) uncommon sites. 110,121,133,134 In a review of 929 cases of mucormycosis, most common sites of involvement were sinuses (39%), pulmonary (24%), skin/soft tissue infection (SSTI) (19%), and disseminated (23%).…”

“…5). Invasion of pulmonary vessels may result in necrotizing pneumonia, 38 pulmonary arterial aneurysms, 193 and hemoptysis (even fatal). 114,194 Invasive ulcerative tracheobronchitis at the anastomotic site in LTRs is much more common with Aspergillus spp., but Rhizopus tracheobronchitis has been described.…”

“…195 Overall mortality with pulmonary mucormycosis ranges from 50 to 70%, but rates exceed 90% with extrathoracic dissemination. 38,114 For localized pulmonary mucormycosis, surgical resection (coupled with medical therapy) may be lifesaving. 193 Invasive tracheobronchitis due to Mucorales or other molds in mechanically ventilated patients in intensive care units (ICUs) with overall mortality of 93.5% was recently described.…”