Asian Cardiovasc Thorac Ann 2000;8:183-185
© 2000 Asia Publishing EXchange Pte Ltd
Combined Medical and Surgical Treatment for Pulmonary Mucormycosis
Tohru Mawatari, MD,
Masanori Nakamura, MD,
Tokuo Koshino, MD,3,
Katsuyuki Kusajima, MD,3,
Tomio Abe, MD,3,
Kazunori Tsunematsu, MD,1,
Hiroyuki Sugawara, MD,1,
Isao Takeya,2
Division of Thoracic Surgery
1 Department of Respiratory Medicine
2 Department of Laboratory
Tomakomai Prefectural Hospital
Tomakomai, Japan
3 Department of Thoracic and Cardiovascular Surgery
Sapporo Medical University School of Medicine
Sapporo, Japan
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For reprint information contact: Tohru Mawatari, MD Tel: 81 11 611 2111 Ext. 3312 Fax: 81 11 613 7318 email mawatari{at}sapmed.ac.jp Department of Thoracic and Cardiovascular Surgery, Sapporo Medical University School of Medicine, South 1 West 16, Chuo-ku, Sapporo 060-8556, Japan.
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Abstract
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A 40-year-old woman with primary pulmonary mucormycosis was successfully treated with combination therapy involving administration of amphotericin B and surgery.
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Introduction
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Mucor is a genus of fungi of the family Mucoraceae, order Mucorales, which forms delicate white tubular filaments and spherical black sporangia; it is broadly distributed in nature. In many cases, mucormycosis occurs as an opportunistic infection in immunosuppressed patients. It affects the nose, brain, lungs, skin, and gastro-intestinal tract. Diagnosis and medical treatment of this disease is difficult, therefore, the mortality rate is high.
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Case Report
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A 40-year-old woman was investigated for bloody sputa and hemoptysis. Physical examination and hematology showed no abnormality. Chest radiography indicated a nodule in the upper field of the right lung (Figure 1
). Computed tomography demonstrated a nodule with cavity formation in right S2 (anterior segment of upper lobe, according to nomenclature of Boyden1) as shown in Figure 2. Bronchofiberscopy revealed inflammation in right B2 and the distal side of the superior truncus bronchialis. Mucor fungus was detected by microscopy in lavage from the right superior truncus bronchialis. Based on these findings, the patient was diagnosed as having pulmonary mucormycosis. Local treatment with 10 mg amphotericin B delivered via the bronchofiberscope was administered at the outpatient clinic once every 2 weeks for 7 months. During this period, no bloody sputum was observed and chest radiography indicated no enlargement of the nodule, but low-grade fever and general malaise gradually progressed so the patient was admitted for further treatment. Local application of amphotericin B was continued at weekly intervals combined with imipenem, cilastatin sodium, and levofloxacin. After 7 weeks, the subjective symptoms had improved and the nodule was slightly smaller. However, full remission was not achieved so it was decided to operate and remove the mucor nidus to obtain a complete cure.
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Figure 1. Chest radiograph showing an abnormal shadow with a nodular appearance in the right upper lung field.
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Prior to surgery, it was confirmed by bronchofiberscopy that there was no inflammation in the right superior truncus bronchialis. A right posterolateral incision was made through the 4th intercostal space. Severe adhesion was found between the right upper lobe, the anterior chest wall, and the middle lobe. There were no abnormal findings in or around the lower lobe. The hilum and mediastinum were not very edematous but the lymph nodes around the bronchus lobaris superior were markedly swollen. The adhesion of the upper lobe and chest wall was dissected but it was not possible to dissect the adhesion between the upper and middle lobes. Part of the hilum pulmonis was dissected and the vessels of the upper lobe were exposed. The vessels were ligated and severed. The bronchi lobaris superior was cut and sutured. The upper lobe and a part of the middle lobe (upper half of S4) were resected. The operation was completed without any problems. The maximum diameter of the resected specimen was 30 mm and a 10-mm cavity was present in the center of the lesion. Histopathology revealed inflammation in the wall of the bronchus and a mass of hyphae (Figures 3A and B). The lymph nodes around the bronchus lobaris superior also showed inflammatory changes. There was no evidence of blood vessel destruction by the lesion.
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Figure 3. Histology of the resected specimen. (A) The wall of the bronchus (arrow) and mass of hyphae (central area); inflammatory cells are seen in the wall of bronchus (hematoxylin and eosin stain, original magnification x40). (B) High magnification of the hyphae shown in A (hematoxylin and eosin stain, original magnification x400).
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Postoperatively, the inflammation subsided over 3 weeks and febricula persisted for a further week. Accordingly, antibiotics were administered for approximately one month postoperatively. The laboratory data and symptoms gradually improved and the patient was discharged from hospital on the 37th postoperative day. She has been followed up regularly for nearly 4 years and no signs of relapse have been detected.
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Discussion
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Mucormycosis accounts for 3.45% to 6.2% of fungal infections.2,3 It occurs mostly in immunosuppressed patients, such as those with severe diabetes mellitus or leukemia, those receiving chemotherapy, and transplant recipients.2,4,5 Hotchi2 reported that secondary mucor-mycosis accounted for 99% of all cases, therefore, primary pulmonary mucormycosis as in our patient, is very rare.
Diagnosis of pulmonary mucormycosis is generally difficult because the radiographic pattern, clinical symptoms, and laboratory data are not specific, thus, the diagnosis is often not obtained until autopsy.2,6,7 According to a previous report, only 44% of patients with pulmonary mucormycosis were diagnosed before death and cases diagnosed by bronchoalveolar lavage, as in our patient, accounted for only 2% of these.8 In this case, the many hyphae found in the lavage indicated that the nidus existed as a localized fungus ball in an area accessible to the fiberscope; diagnosis at this stage allowed early treatment. The in-hospital mortality rate for patients with pulmonary mucormycosis has been reported as 80% and surgery was found to be superior to medical treatment (surgical mortality 11% versus medical mortality 68%).8 Therefore, surgery should be the treatment of first choice, especially in the case of a localized lesion. However, combined medical and surgical therapy should be performed depending on the location and size of the lesion, to reduce the size and make it more amenable to surgical treatment. In this patient, reduction of the nidus was achieved initially by medical treatment because the inflammation was located in the superior truncus bronchialis and the lesion might have remained in the stump after lobectomy.
Amphotericin B is widely accepted as the most effective drug for mucormycosis.5,9 It is usually administered systemically and rarely locally. While it has proved effective when used systemically, severe side effects such as renal dysfunction, liver dysfunction, gastrointestinal symptoms, general malaise, and headache are frequent. When these side effects occur, the administration of amphotericin B must be stopped. However, we observed neither subjective symptoms nor objective signs of side effects in our patient during local treatment with this drug. We have applied local treatment with amphotericin B in 5 patients with localized pulmonary mycosis in the past year and have not observed any severe side effects. Therefore, we think that local treatment is superior in cases of localized mucormycosis with regard to both efficacy and side effects.
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References
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Boyden EA. General survey of bronchial tree. Segmental anatomy of the lungs. New York: McGraw-Hill, 1955: 23–32.
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Hotchi M. Pathology of phycomycosis. Jpn J Med Mycol 1978;19:94–100.
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Koziel H, Koziel MJ. Pulmonary complications of diabetes mellitus. Infect Dis Clin North Am 1995;9:65–96.[Medline]
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Hawkins C, Armstrong D. Fungal infections in the immu-nocompromised host. Clin Haematol 1984;13:599–630.[Medline]
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Senba M, Toda T, Toda Y, Hokama S. A reliable differ-entiation of Mucor from Aspergillus in tissue sections with ultraviolet illumination. Acta Med Nagasaki 1987; 34:147–8.
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Rubin SA, Chaljub G, Winer-Muram HT, Flicker S. Pulmonary zygomycosis: a radiographic and clinical spectrum. J Thorac Imaging 1992;7:85–90.[Medline]
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Tedder M, Spratt JA, Anstadt MP, Hegde SS, Tedder SD, Lowe JE. Pulmonary mucormycosis: results of medical and surgical therapy. Ann Thorac Surg 1994;57:1044–50.[Abstract]
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Valicenti JF Jr, Conte JH. Successful medical manage-ment of pulmonary phycomycosis. South Med J 1980; 73:384–6.[Medline]
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Abstract
Introduction
