Unusual Presentation of Pulmonary Mucormycosis in an Immunocompetent Patient

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1 World Journal of Medical Sciences 14 (2): 47-51, 2017 ISSN IDOSI Publications, 2017 DOI: /idosi.wjms Unusual Presentation of Pulmonary Mucormycosis in an Immunocompetent Patient Muhammad Imran Khan and Maria Tanvir Department of Radiology, Fatima Jinnah Medical University, Lahore, Pakistan Abstract: A fifty-eight yearsold immune-competent man presented to medical department with history of fever, chest pain, cough andweight loss from last 15 days. He has been smoking for40 years.his X-ray chest showed large lung field opacitiesbilaterally and was referred to us for CT Chest with IV contrast.ct chest revealed few large cavitieshaving nodular thick walls, present in both lungs. Since there was no previous history of immune deficiency excluding probability of primary fungal disease while tuberculosis remained a differential and CT features were also somewhat suspicious for primary or metastatic neoplastic disease. CT guided trucut-biopsy was performed. Histopathology showed necrotizing granulomatous inflammation with fungal hyphae which were non-septated and ribbon shaped, favoring pulmonarymucormycosis.patient was put on Amphotericin B(Anti-fungal) therapy. During his hospital stay, patient developed severe backache and was again referred to us for MRI lumbosacral spine which showed disco vertebral erosive disease with multiple intra and extraspinalabscesses that was suggestive forpott s spine. Patient was put on anti-tuberculoustherapy as well. After 2 months of therapy, patient re-appeared for follow up CT Chest and it displayed marked resolution of pulmonary disease process indicative of prompt response totherapy. It may also be assumed that pulmonary mucormycosis was secondarily infesting tuberculous cavities, although there was no histological evidence of acid fast bacilli in acquired samples, previous history of tuberculosis was also lacking and disease presentation was rather acute. Key words: Mucormycosis Immune-Competent Amphotericin B Antituberculous CT Guided Biopsy INTRODUCTION pulmonary mucormycosis has increased with the development of modern medicine. Numerous predisposing Pulmonary mucormycosis is an opportunistic clinical factors have been described, including pulmonary fungal infection from a fungus belonging to uncontrolled diabetes mellitus, diabetic ketoacidosis, the order Mucorale [1]. Pulmonary mucormycosis is a chemotherapy, hematological malignancies (Leukemia and relatively uncommon infection that occurs mostly in lymphoma), immunosuppressive therapy, acquired or immunocompromised persons. The first case of congenital neutropenia, antibiotic therapy, metabolic pulmonary mucormycosis was described in 1876 by acidosis due to chronic salicylate poisoning, elastoplast Furbringer [2]. Mucorales fungi are ubiquitous, bandages, renal failure, a prolonged post-operative saprophytic andnon fastidious fungi located in soil or course, solid tumors, solid organ transplantation, decaying organic matter, with three genera that are known agammaglobulinemia and burns [6-8]. Only 6.25% patients to be human pathogens, namely, Rhizopus, Absidia and do not have any underlying risk factor [9, 10]. This study Mucor. The optimal temperature for growth is 28 to 30 C described a case of an immunocompetent patient who was under aerobic conditions, with an incubation period of unsuspected for pulmonary mucormycosis and he also 2 to 5 days. Incubation begins with inhalation of the had undiscovered ongoing lumbar spondylodiscitis. spores or their direct inoculation into abraded skin [3]. The estimated incidence of the disease is 1.7 cases Case Report: A previously healthy 58 years old male was per million people per year in the United States [4]. In a presented to medical department of Sir Ganga Ram review of 116 cases of mucormycosis, 22% were hospital, Lahore, Pakistan with 15 days history of pulmonary mucormycosis[5]. However, the incidence of fever, chest pain and weight loss. There was no history Corresponding Author: Dr. Muhammad Imran Khan, Department of Radiology, Fatima Jinnah Medical University, Lahore, Pakistan. 47

2 Fig. 1: Chest X Ray PA view of 58 years old male, obvious lung cavities bilaterally Fig. 2: CT Chest with IV contrastexhibits thick wall cavities in right upper lobe approaching pleural surface laterally. Another similar characteristic largecavityin lingular segment of left lobe measuring 8.4 x 5.3 x10 cm. Both these lesions have thicknodular walls. ofhemoptysis, chills or night sweats. The patient was 6 cm in size, was approaching pleural surface laterally; smokingfor 40 years. On physical examination he was another similar characteristic large cavity was noted in afebrile with normal pulse rate and blood pressure. lingular segment of left upper lobe measuring 8x10 cm. His lab investigations revealed normal renal, hepatic Both these lesions showed nodular wallshaving and serum electrolyte profile. He was negative for asymmetric thickness measuring upto 18mm in Hepatitis B/C and HIV 1-2. diameter.findings of bilateral upper lung lobe His Chest X-ray (Fig. 1) was obtained which showed cavitieshaving nodular mural thickening were suspicious pulmonary opacities with central cavitation present for primary or secondary malignancy with probability bilaterally and was advised CT Chest with IV contrast. His of tuberculosis. Fungal disease was a remote possibility CT scan(fig. 2) was performed which showed thick walled and was unsuspected since history of this illness was pulmonary cavities in either lung. One in right upper lobe, short and patient was well in health before onset of the 48

3 a b c Fig. 3(a, b, c): Sagittal T1 and T2 weighted sequences, axial T1W with contrast. T1 intermediate, T2 hyperintenseintraspinalextrathecallesion spanning along posterior border of L5 vertebra. Pockets of fluid in bilateral paraspinal muscles and in right psoas muscle are also obvious, more marked on left side from L2 to L5 levels suggestive of multiple intra and extra spinal collections/abscesses. Fig. 4: Follow up CT showing significant reduction in disease process, size of largest cavityin lingular segment of left upper lobe decreasing from 8.4x5.3x10 cm to 4.7x4.0x6.0cm along with thinning of cavity walls symptoms. Later on, CT guided Tru Cut-biopsy was was immunocompetent. Patient was put on anti-fungal performed in our department of Radiology, Sir Ganga Ram therapy, Amphotericin B. Patients clinical symptoms Hospital using 18G needle, obtaining samples from the started to settle. During his stay at hospital he complained nodular wall of larger left lung cavity. Samples were sent of backache radiating to both legs. His physician advised to Pathology department, which came out to be MRI Lumbosacral spine. His MRI (Fig. 3 a, b, c) showed necrotizing granulomatous inflammation with fungal multiple intra and extra spinal collections/abscesses, hyphae which were fragmented and non septatedribbon suspicion of tuberculosis was raised and patient was put shaped, favoring Mucormycosis. Pulmonary on anti-tuberculous therapy too. After two months of mucormycosis was a rare diagnosis in this case as patient anti-fungal and one month of anti-tuberculoustherapy, 49

4 follow up contrast enhanced CT Chest (Fig. 4) was REFERENCES acquired which displayed moderate interval reduction 1. Bennett, J.E., R. Dolin and M.J. Blaser, in size of cavities, from8x10 cm to 4x6.0cm of the main th Principles and Practice of Infectious Diseases. 8 lesion on left side. Significant size reduction and edition, Saunders. improved clinicalsymptoms proved the efficacy of 2. Fürbringer, P., Observations on pulmonary anti-fungaltherapy, Amphotericin B in this case of acute mucormycosis in humans. Virchows Arch. Path. mucormycosis. It may be speculated that pulmonary Anat. Physiol Klin Med., 1876(66): doi: mucormycosis was secondarily infesting pulmonary /BF tuberculous cavities, although there was no histological 3. Spellberg, B., Edwards J. Jr and A. Ibrahim, evidence of acid fast bacilli in acquired samples, previous Novel perspectives on mucormycosis: history of tuberculosis was also lacking and disease pathophysiology, presentation and management. Clin presentation was rather acute. Microbiol. Rev., 18(3): Garg, R., R.S. Marak, S.K. Verma, J. Singh, Sanjay DISCUSSION and R. Prasad, Pulmonary mucormycosis mimicking as pulmonary tuberculosis: A case Pulmonarymucormycosis is uncommon but a life- report. Lung India, 25: doi: /0970- threatening situation. Pulmonary mucormycosis has high morbidity and mortality.mucormycosis should be 5. Aboutanos, M.B., M. Joshi and T.M. Scalea, considered in the differential diagnosis of cavitatinglung Isolated pulmonary mucormycosis in a patient with lesions in patients with poorly controlled diabetes or multiple injuries: A case presentation and review of other immunosuppressed states.to prevent delays in the literature. J. Trauma, 54: doi: treatment and improve the likelihood of survival, early /01.TA B. diagnosis is imperative. Early surgical resection and 6. Bigby, T.D., M.L. Serota, L.M. Tierney, Jr, M.A. amphotericin antifungal therapy are considered the Matthay, Clinical spectrum of pulmonary standard treatment.future prospective studies are needed mucormycosis. Chest, 89: doi: to evaluate outcomes in pulmonary mucormycosis treated /chest with early surgical resection and combination of 7. Muqeetadnan, M., A. Rahman, S. Amer, S. Nusrat, amphotericin B and echinocandins [11]. S. Hassan and S. Hashmi, Pulmonary Diagnosis can be particularly challenging in part mucormycosis: An emerging infection. Case Rep. because of its relative rarity. On chest imaging, pulmonary Pulmonol., mucormycosis may present with focal consolidation, 8. Mohammadi, A., A. Mehdizadeh, M. Ghasemi-Rad, lung masses, pleural effusions, or multiple nodules [12]. H. Habibpour and A. Esmaeli, Pulmonary Direct histological examination of the tissue biopsy mucormycosis in patients with diabetic ketoacidosis: remains the gold standard for diagnosis. The A case report and review of literature. TuberkToraks, histopathological findings reveal irregular broad 60: doi: /tt nonseptated hyphae and spores [13]. 9. Von Scheven, R., P. Lebiedz, T. Spieker, A. Uekoetter, W.E. Berdel and T. Kessler, Fulminant CONCLUSIONS invasive pulmonary mucormycosis with Rhizopusoryzae in a patient with severe aplastic This reported case is unique in a sense that anaemia and common variable immunodeficiency. Pulmonary Mucormycosis is seen in immunocompromised Mycoses, 55: e32-e35. patients mostly. In the case reported, patient was 10. Roden, M.M., T.E. Zaoutis, W.L. Buchanan, immunocompetent, history was short and there was no T.A. Knudsen, T.A. Sarkisova, R.L. Schaufele, M. prevailing illness at the time of presentation, so Sein, T. Sein, C.C. Chiou, J.H. Chu, Dimitrios P. mucormycosis was not considered in its differentials. But Kontoyiannis and Thomas J. Walsh, the biopsy proved it to be fungal infection, Pulmonary Epidemiology and outcome of zygomycosis: A Mucormycosis and there was excellent clinical and review of 929 reported cases. Clin Infect Dis., 41: 634- radiological response after Anti-fungal therapy doi: /

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