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Year : 2003  |  Volume : 5  |  Issue : 1  |  Page : 38-41

Invasive aspergillosis of the cervical region and mediastinum - Case report and review of literature

Division of ENT, King Khalid National Guard Hospital, Jeddah, Saudi Arabia

Date of Web Publication11-Jul-2020

Correspondence Address:
M.D., FRCS(C), FACS Essam Al-Zimaiti
Division of ENT, King Khalid National Guard Hospital P. O. Box 9515 Jeddah, 21423
Saudi Arabia
M.D., FRCS(C) Saad Al-Muhayawi
Division of ENT, King Khalid National Guard Hospital P. O. Box 9515 Jeddah, 21423
Saudi Arabia
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1319-8491.289564

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Aspergillus species are ubiquitous in the environment worldwide. They can be found in the soil, water and vegetation. Non-invasive aspergillus lesions maydevelop in healthy paranasal sinuses or in pre-existing pulmonmary cavities. However, invasive aspergillosis (IA) is primarily an infection of severely immunocompromised patients and one of the most life-threatening conditions in this group of patients, with very high fatality rate. However, in immunocompetent persons, fulminant invasion of tissues is very rare. We report a case of a 30-year-old Saudi lady who developed invasive aspergillosis of the cervicalregion extending into the mediastinum and the vertebral foramina. This unusual infection was produced simultaneously by two species of Aspergillus i.e. A.fumigatus and A. niger. She was treated with a combination of amphotericin B i.v and itraconazole orally for 6 months. She responded very well to the combination therapy and made a remarkable recovery. Treatment options of this fatal infection are briefly reviewed and discussed.

Keywords: Invasive Aspergillosis; Treatment; Amphotericin B; Itraconazole; Voriconazole

How to cite this article:
Al-Zimaiti E, Al-Muhayawi S. Invasive aspergillosis of the cervical region and mediastinum - Case report and review of literature. Saudi J Otorhinolaryngol Head Neck Surg 2003;5:38-41

How to cite this URL:
Al-Zimaiti E, Al-Muhayawi S. Invasive aspergillosis of the cervical region and mediastinum - Case report and review of literature. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2003 [cited 2022 Dec 8];5:38-41. Available from: https://www.sjohns.org/text.asp?2003/5/1/38/289564

  Introduction: Top

Aspergillus species are ubiquitous in the environment worldwide. They can be found in the soil, water and vegetation. Nearly 200 species of Aspergillus have been identified but less than 20 species have been found to be pathogenic for humans. The most common species causing human infections (Aspergillosis) are A. fumigatus , A. flavus and A, niger [1],[2], Since Aspergillus spp. are primarily respiratory pathogens, most infections involve the paranasal sinuses and the lungs, although primary skin infections can occur both in healthy and immucompromised hosts [2], Noninvasive aspergillomas may develop in healthy paranasal sinuses or in pre-existing pulmonmary cavities. However, invasive aspergillosis (IA) is primarily an infection of severely immunocompromised patients and one of the most life-threatening conditions in this group of patients. It occurs most commonly in patients with haematological malignancies, malignant lymphomas, solid tumors, organ transplant recipients and drug-induced immunosuppression. IA is associated with a fatality rate approaching 100% [2],[3],[4],[5],[6].

We describe a case of invasive aspergillosis of the cervical region and mediastinum in an immunocompetent 30 year old patient.

  Case Report: Top

The patient was a 30-year-old Saudi lady who delivered a baby two weeks before attending our hospital. She presented with a mass on the left side of the neck, which has been present for the past one year and had progressively became larger. Three months prior to presentation, she suddenly developed pain in her left upper limb associated with weakness and two months later she developed hoarseness of her voice. She had noticed gradual loss of weight and appetite over the preceding two months. On examination, she looked ill with ptosis of the left eye. The respiratory and cardiovascular systems were essentially normal. There was no organomegaly. A hard fixed mass was palpable over the lower part of the left side of neck and extending from the region of the left lobe of the thyroid to the anterior border of the trapezius muscles. It was about 3cm wide and 15 cm long and extending into the sternomastoid muscle. The lower border could not be reached, but produced dullness over the sternum. Although there were no visible pulsations, the trachea has been shifted to the right side. There were normal pulsations of the carotid and radial arteries, but weakness of the left upper limb with wasting of the small muscles of the hand was noticed. Ear, nose and throat examination showed left vocal cord paralysis. The sinuses were normal on examination. The C-T scan showed a huge mass occupying the left side of the neck extending to the mediastinum, with the thyroid displaced anteriorly and towards the right. There was an extension of the mass into the spinal canal, with slight displacement of the spinal cord. [Figure 1]. Routine haematological investigations were normal. A fine needle aspiration of the left lobe of the thyroid was carried out and it showed merely thy- roditis. A preliminary diagnosis of a metastatic malignant tumour of undetermined primary lesion was made. She was booked for panendoscopy and biopsy of the left cervical mass. At operation, a very large indurated mass measuring 6cm by 1 Ocm was discovered. The mass was gritty and hard in consistency and could not be dissected from the carotid sheath as it was stuck to theposterior structures of the neck. Biopsies were obtained for histological and microbiological investigations. The histological examination showed chronic inflammation, fibrosis, and necrotizing granulomata, with no evidence of malignancy seen. Histochemical stains of the biopsy material revealed irregular non- septate hyphae with variable diameters within the granulomata. Ziehl Neelsen stain for Mycobacteria was negative. It was concluded that this was a chronic inflammation due to fungal infection. Microbiological investigations of the biopsy material by wet smear and culture revealed Aspergillus fumigatus and A. niger [Figure 2]. Sputum examination by Gram stain did not reveal any hyphae suggestive of Apsergillus. Ziehl Neelsen stain of the sputum did not show the presence of any acid- fast bacilli, A diagnosis of invasive aspergillosis with A, fumigfatus and A. niger was recorded.
Figure 1: C-T scan of the cervical region showing a hug mass occupyinn the left side of the neck infiltrating into the mediastinum

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Figure 2: PAS and Silver showing fungal elements with non-septate hyphas in an area of inflammation, fibrosis and necrotizing granuloma

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  Management: Top

The patient was placed on a regime of amphotericin 1 mg/kg/day iv for one month and itraconazole 400m mg daily orally for 6 months. She showed considerable improvement and was discharged home after 6 weeks of treatment in the hospital and given an appointment for 2 months for outpatient review. Six months after discharge, the mass in the neck had almost completely resolved and the movement and power of the left hand had improved. The CT scan performed after 12 months of starting therapy showed that the mass had completely resolved, with no mass effect on the oesophagus, trachea or thyroid. However the residual mass was still engulfing the left internal carotid artery. There was complete regression of the intraforaminal extension at C7 and T1 levels. Her ptosis has improved considerably. Overall, she made a spectacular recovery.

  Discussion: Top

Aspergillosis refers to the broad range of disease states whose aetiologic agents are members of the genus Aspergillus. They are ubiquitous soil saprophytes and are found worldwide. Aspergillus can cause a variety of illnesses such as simple sputum colonization, hypersensitivity pneumonitis, allergic rhinosinusitis, pulmonary aspergillomas (fungus balls) and invasive aspergillosis (IA) [4],[7],[8],[9]. Aspergillosis may be acquired by inhalation of airborne spores in the proper host, such as the compromised host. IA has been increasingly reported as an opportunistic pathogen in the compromised host, second only to Candida spp. in this setting. [4],[7], Patients with leukemia and lymphomas as well as transplant recipients are particularly prone to infection. Similarly, patients with other malignant diseases with leukopenia or on immunosuppressive agents, corticosteroids, or antibiotics are also at increased risk, so are patients with chronic granulomatous disease, who are particularly susceptible because of their granulocytic defect. However, pulmonary involvement is present in 90- 95% of cases and dissemination occurs in about 25%. [4],[7].

The pathology of IA depends on the host.In immunocompromised patients, vascular invasion is paramount, leading to infarction necrosis, edema, and hemorrhage in distal tissues. Hyphae are abundant, and forming radially branching clusters in the tissue. In contrast vascular invasion is not seen, and hyphae are sparse in tissues from patients with chronic granulomatous disease. The hyphae of Aspergillus are about 2-4 pm wide, frequently septate and dichotomously branched [1]. In rapidly progressing infections, the hyphae tend to be of even diameter. In sites of indolent disease, hyphae may have bulbous widened area [1].

The case reported here is unique for the following reasons. Firstly, this patient was immunocompetent and she was not on immunosuppressive medication. Secondly, there was no history of corticosteroid therapy. Thirdly, there was no evidence of any of the predisposing factors for the development of Aspergillosis such as inhalation of spores leading to pulmonary infection or living in a farm, where decaying vegetation often contain large amounts of spores. Fourthly, there was no evidence of allergic manifestation of aspergillosis such as hypersensitivity pneumonia or allergic bronchopulmonary aspergillosis or paranasal sinusitis. Finally two different species of Aspergillus were isolated from the biopsy material i.e. A. fumigatus and A. niger. The only probable explanation for this patientis invasive infection was that her immunity might have been compromised by her pregnancy as well as the double infection by two strains of Aspergillus simultaneously.

There are increasing reports of the various manifestations of Aspergillosis in Saudi Arabia. The most common manifestation reported has been the Allergic fungal rhinosinusitis [8],[9],[10],[11],[12],[13]. The later condition has been noted to be common in the Sudan and Saudi Arabia. IA can manifest as pulmonary or extrapulmonary aspergillosis. The pulmonary effects of IA usually manifest a necrotizing bronchopneumonia, while extrapulmonary dissemination is found in approximately 10-25% of patients at autopsy, The lesions can be found in the brain, gastrointestinal tract, liver, renal, myocardial, endocardial and paranasal infections [7], IA is a fulminating disease that carries a high fatality rate. Survival of the patient depends on early diagnosis and therapy while the prognosis is partially dependent on the underlying disease [7], Intravenous amphothericn B is the drug of choice for IA. Synergy in vitro has been reported against some species of Aspergillus when amphotericin is combined with rifampicin. Similarly variable results have been reported when amphotericin B is combined with flucytosine [14]. The optimal duration of amphotericin B treatment is unknown, but cessation of therapy must be guided by the clinical response of the patient. Commonly, high doses of amphotericin B are used in IA, i.e. 1 mg/kg/day and in patients not responding, doses as high as 1.5 mg/kg/day has been used. Liposomal amphotericin B has been shown to be efficacious and safe in the management of severe infections, particularly in patients with severe renal impairment and can be used at a dose of 2 mg/kg daily, without renal toxicity [15],[16],[17],[18], More recently, voriconazole, a wide-spectrum triazole antifungal agent active in vitro against Aspergillus species, with an MIC 0.4 mg/L has been shown to be safe and efficacious in acute IA [19], [20],[21],[22]. It has the advantage that it can be given orally and intravenously making switch therapy possible. The recommended protocol for initial therapy is two intravenous loading doses of 6 mg/kg at 12 hourly intervals for 6-27 days followed by 3 mg/kg at 12 hourly intervals, then oral therapy of 200 mg b.i.d. for 4-24 weeks [22]. Our patient responded very well to the combination of amphotericin B and itraconazole.

  References Top

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Patel PJ, Kolawole TM, Malabarey TM, Hulailah A, Hamid F, Chakaki M. CT findings in paranasal aspergillosis. Clin Radiol 1992;45(5):319-321.  Back to cited text no. 12
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Verweij PE, Donnelly JP. Kullberg BJ, Meis JFGM, de Pauw BE. Amphotericn B versus amphotericin B plus 5-flucytosine : poor results in the treatmenwt of proven systemic mycoses in neutropenic patients. Infection 1994;22 :81-85.  Back to cited text no. 14
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Denning DW , Ribaud P, Milpied N, Caillot D, Herbrecht R, Thiel E, Haas A, Ruhnke M, Lode H. Efficacy and Safety of Voriconazole in the Treatment of Acute Invasive Aspergillosis. Clin Infect Dis 2002; 34:563-571.  Back to cited text no. 19
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Verweij PE, Mensink M, Rijs AJMM. Donnelly JP, Meis JFGM, Denning DW. In vitro activities of amphotericin B, itraconazole and voriconazole against 150 clinical and environmental Aspergillus fumigatus isolates. J Antimicrob Chemother 1998; 42 :389-392.  Back to cited text no. 21
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  [Figure 1], [Figure 2]


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