Intracranial Meningioma in a Patient with ABPA: A Case Report and Literature Review | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Afro-Egyptian Journal of Infectious and Endemic Diseases | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Article 15, Volume 15, Issue 3, September 2025, Page 343-353 PDF (538.93 K) | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Document Type: Case report | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
DOI: 10.21608/aeji.2025.388285.1481 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
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Authors | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Sheetal Goenka![]() ![]() ![]() ![]() ![]() ![]() ![]() | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Department of Microbiology, Govind Ballabh Pant Institute of Postgraduate Medical Education and Research (GIPMER) New Delhi, India. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Abstract | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Meningiomas are the most common primary intracranial tumours, accounting for about 36% of central nervous system neoplasms. Arising from arachnoid cap cells, they are typically benign, slow-growing, and often found incidentally, though symptoms may occur from compression of nearby neural structures. We present a case of intracranial meningioma in a 25-year-old female with allergic bronchopulmonary aspergillosis (ABPA) caused by Aspergillus terreus. The patient presented with holocranial headache, diminished right eye vision, and other neurological symptoms. Radiological investigations revealed a right temporal space-occupying lesion with characteristic features of meningioma. The patient underwent right temporal craniotomy with tumour decompression. Histopathological examination confirmed the diagnosis of meningioma. This case highlights the importance of considering meningioma in the differential diagnosis of CNS lesions, even in patients with concurrent fungal disease in other organ systems. Additionally, it draws attention to Aspergillus terreus as a causative agent of ABPA, which is relatively uncommon compared to Aspergillus fumigatus. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
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Intracranial meningioma; Allergic bronchopulmonary aspergillosis; Craniotomy; Aspergillus terreus; Atypical ABPA | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
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Introduction Meningiomas are the most common primary intracranial tumours, accounting for approximately 36% of all central nervous system tumours [1]. They are typically benign, slow-growing neoplasms that arise from the arachnoid cap cells of the meninges. Most meningiomas are asymptomatic and discovered incidentally, but they can cause symptoms due to compression of adjacent neural structures. Here, we present a case of intracranial meningioma in a young female with concurrent allergic bronchopulmonary aspergillosis (ABPA), an immunological disorder characterized by hypersensitivity to Aspergillus antigens. While Aspergillus fumigatus is the most common cause of ABPA worldwide, our patient presented with ABPA due to Aspergillus (A.) terreus, which is relatively uncommon but increasingly recognized as a causative agent of ABPA [2]. A. terreus accounts for approximately 3-5% of ABPA cases and is notable for its intrinsic resistance to amphotericin B, which may influence treatment decisions [3,4]. Case presentation A 25-year-old woman presented to the neurosurgery department at GB Pant Hospital in New Delhi with a six-month history of diffuse headache, gradually worsening vision in her right eye, dizziness, and nausea. She reported no episodes of loss of consciousness, vomiting, or seizures. The patient had a 3-year history of recurrent respiratory symptoms, including cough, wheezing, and dyspnoea, previously diagnosed as allergic asthma. She had no other known comorbidities or history of immunosuppressive therapy. On physical examination, the patient was afebrile with stable vital signs (pulse rate 84/min, blood pressure 110/70 mmHg). Neurological examination revealed normal higher mental functions, intact cranial nerves, normal muscle tone and power in all limbs, and preserved deep tendon reflexes. There were no signs of meningeal irritation. Respiratory examination revealed bilateral scattered rhonchi and occasional crackles. Diagnostic Workup
While the total serum IgE level (365.5 IU/mL) was elevated above normal range, it was below the typical threshold (>1000 IU/mL) for classical ABPA according to ISHAM criteria [5]. This may represent an early or atypical presentation of ABPA, sometimes seen with non-fumigatus species. The strong specific IgE response against A. terreus (8.7 KU/mL) and positive precipitins, along with radiological findings, supported the diagnosis of ABPA despite the lower total IgE.
Sputum examination:
After 3 days, colonies appeared compact, velvety, and cinnamon-brown to buff-coloured The reverse side of the colonies displayed a pale yellow to brown hue.
Microscopy of the culture revealed – Hyaline, septate hyphae Conidiophores smooth-walled, becoming roughened near the vesicle Biseriate conidial heads with compact columnar arrangement Vesicles were dome-shaped, measuring 10–16 μm in diameter, with metulae and phialides covering the upper half of the vesicle. Importantly, characteristic globose to ellipsoidal aleurioconidia (accessory conidia) were observed arising laterally from the hyphae, confirming A. terreus identification [6]. While morphological characteristics strongly suggested A. terreus, molecular identification techniques such as ITS region sequencing or MALDI-TOF mass spectrometry would have provided definitive species confirmation, which is recommended for non-fumigatus Aspergillus species with clinical significance [7].
Antifungal susceptibility testing should have been performed on the A. terreus isolate according to CLSI or EUCAST guidelines to determine minimum inhibitory concentrations (MICs) for various antifungals, which would have guided more precise therapy. This is particularly important for A. terreus, which can show variable susceptibility patterns to triazoles [8]. A detailed environmental history was obtained to identify potential sources of A. terreus exposure. The patient reported having multiple potted plants at home and periodically engaging in gardening activities, which are known risk factors for exposure to A. terreus, as it is predominantly found in soil environments [3].
DIAGNOSIS Based on the clinical, laboratory, mycological, and radiological findings, the following diagnoses were established: Right temporal meningioma (WHO grade I) Allergic bronchopulmonary aspergillosis (ABPA-CB stage) according to ISHAM working group criteria, with sensitization predominantly to Aspergillus terreus. MANAGEMENT AND HOSPITAL COURSE Preoperative Management: Prior to neurosurgical intervention, the patient's ABPA was stabilized with:
It is important to note that in vitro susceptibility patterns do not always correlate with clinical response, particularly for A. terreus, which can demonstrate paradoxical growth effects in the presence of certain antifungals despite apparent in vitro susceptibility [9]. Surgical Management: The patient underwent right temporal craniotomy with tumour decompression. Intraoperative findings included tense dura, bulging brain, and a firm, moderately vascular extra axial mass arising from the temporal base with arachnoid invasion. A Simpson Grade II resection was performed, indicating complete removal of the tumour along with coagulation of its dural attachment. [10]. Histopathological Findings: Histopathological examination of the excised tissue confirmed the diagnosis of meningioma (WHO grade I), demonstrating whorled nests of meningothelial cells with intranuclear pseudo inclusions and psammoma bodies. Immunohistochemistry demonstrated positive expression of epithelial membrane antigen (EMA) and vimentin, supporting the diagnosis of meningioma. The Ki-67 labelling index was found to be 2%, suggesting low cellular proliferation. Postoperative Management: Following surgery, the patient was initiated on the following treatments:
Continued ABPA management with:
HOSPITAL COURSE The patient's hospital stay was uneventful. Regular dressings were performed, and stitch line remained healthy. At the time of discharge (day 6 post-surgery), the patient was vitally stable, taking oral medications, with a healthy stitch line and no new neurological deficits. Serial serum IgE levels were planned for monitoring ABPA response to therapy. Quantitative culture techniques to assess fungal burden, such as colony-forming unit counts from sputum samples, were initiated to provide objective metrics for monitoring treatment response beyond clinical symptoms and IgE levels [11]. FOLLOW-UP and OUTCOMES The patient was discharged with instructions to follow up in the neurosurgery outpatient department after 7 days and again after 2 weeks. At the first follow-up visit, the surgical site appeared healthy with no signs of infection. The patient reported significant improvement in headache but continued to have mild visual disturbances in the right eye. At the 1-month follow-up visit, contrast-enhanced MRI brain showed postoperative changes with no evidence of residual or recurrent tumour. Total serum IgE had decreased to 210 IU/mL, indicating good response to ABPA therapy. Pulmonary function tests showed improvement with FEV1 increasing to 78% of predicted. At the 3-month follow-up, the patient demonstrated marked clinical improvement with resolution of headache and partial improvement in right eye vision. The ABPA was well-controlled with tapering doses of oral prednisolone (now at 5 mg/day), continued inhaled corticosteroids, and completion of voriconazole therapy. Repeat sputum culture showed no growth of Aspergillus terreus, suggesting successful clearance of the fungus. At 6 months, the patient underwent comprehensive evaluation at a pulmonology center for management of her ABPA. Her pulmonary symptoms were well-controlled with inhaled corticosteroids and periodic courses of oral prednisone during exacerbations. Total serum IgE had decreased to 180 IU/mL, and chest imaging showed improvement in bronchiectasis and clearing of mucoid impactions. At the 12-month follow-up, the patient remained clinically stable with no evidence of tumour recurrence. MRI demonstrated postoperative changes with no enhancing lesion. Pulmonary function tests showed FEV1 at 82% of predicted. The patient was advised to continue annual MRI surveillance for the next 5 years, then biennial surveillance thereafter, which is the standard follow-up protocol for completely resected WHO grade I meningiomas [12]. For ABPA management, a maintenance regimen of inhaled corticosteroids and as-needed short courses of systemic corticosteroids during exacerbations was recommended, with quarterly monitoring of total serum IgE levels. Discussion Meningioma Considerations Meningiomas are the most common primary intracranial tumours, with an annual incidence of approximately 7.8 per 100,000 individuals [1]. They are more common in females, with a female-to-male ratio of approximately 2:1. The radiological findings in this case were characteristic of meningioma, including a dural-based, extra-axial mass with homogeneous enhancement and dural tail sign. These features are highly suggestive of meningioma and help differentiate it from other intracranial lesions. The management of meningiomas depends on various factors, including size, location, symptoms, and patient characteristics. Surgical resection is the treatment of choice for symptomatic meningiomas, as was done in our patient. The extent of resection is an important predictor of recurrence, with Simpson grade I resection (complete removal of tumour, dural attachment, and involved bone) associated with the lowest recurrence rates [1 0]. ABPA Considerations Allergic bronchopulmonary aspergillosis (ABPA) is classified based on the criteria established by the International Society for Human and Animal Mycology (ISHAM) working group into the following categories:
Our patient presented with ABPA-CB, characterized by the presence of central bronchiectasis on chest imaging. The strongest skin test reaction to A. terreus (8×6 mm) which, combined with its isolation from the sputum culture, is particularly significant—especially considering that ABPA is usually associated with A. fumigatus. Aspergillus terreus ABPA: Special Considerations Aspergillus terreus is an uncommon cause of ABPA, responsible for approximately 3-5% of cases globally [2]. It has several distinctive features that merit special consideration:
The management of ABPA typically includes two key therapeutic strategies:
Our patient was treated with a combination of oral prednisolone and voriconazole, which is consistent with current guidelines adapted for A. terreus [15]. Voriconazole has demonstrated efficacy against A. terreus and may also provide a steroid-sparing benefit in the management of ABPA. The monitoring of treatment response using total serum IgE levels is standard practice, with a 35-50% reduction from baseline indicating a good response to therapy [5]. Concurrent Management Considerations It is noteworthy that our patient had normal CD3+ and CD4+ T-cell counts, indicating intact cellular immunity. This is important because immunocompromised states can be associated with various intracranial pathologies, including opportunistic infections and certain malignancies. The coexistence of ABPA and meningioma in our patient appears to be coincidental, as there is no established pathophysiological link between these two conditions. However, several management considerations were important in this case:
Radiological Considerations The radiological features in this case were initially suggestive of meningioma, as is commonly seen with dural-based lesions. The presence of dural tail sign, homogeneous enhancement, and broad-based attachment to the sphenoid wing are characteristic of meningiomas [16]. Meningiomas typically appear as well-defined, extra-axial masses that are isointense to slightly hypointense on T1-weighted MRI sequences and isointense to slightly hyperintense on T2-weighted sequences. These lesions show uniform enhancement following contrast administration and frequently exhibit a dural tail sign, characterized by thickening and enhancement of the dura adjacent to the tumour. These features were present in our patient's imaging studies, supporting the diagnosis of meningioma. While the presence of paranasal sinus involvement might raise suspicion for invasive processes like fungal infections or malignancies, in this case, it represented extension of the meningioma rather than a separate pathological process. The absence of bone erosion and the presence of a well-defined tumour margin further supported the diagnosis of meningioma over invasive processes. For ABPA, the chest imaging demonstrated characteristic findings of bilateral bronchiectasis and high-attenuation mucus plugging, creating the classic "finger-in-glove" appearance. This radiological pattern is highly suggestive of ABPA and, when combined with clinical, mycological, and immunological findings, is diagnostic. Histopathological Considerations Histopathological examination is the gold standard for diagnosing meningiomas. WHO grade I meningiomas, the most common type, demonstrate various histological patterns, including meningothelial, fibrous, transitional, psammomatous, and angiomatous variants. Our patient's tumour showed features of a meningothelial meningioma with whorled nests of cells and occasional psammoma bodies. Immunohistochemical staining showed positivity for epithelial membrane antigen (EMA) and vimentin, which helps differentiate meningiomas from other dural-based lesions. The absence of atypical features such as increased mitotic activity, brain invasion, or necrosis classified our patient's tumour as a WHO grade I meningioma, which generally has a favourable prognosis. MANAGEMENT APPROACH Meningioma Management: Management of intracranial meningiomas requires a multidisciplinary approach involving neurosurgery, neuroradiology, and sometimes radiation oncology. The principles of management include:
In our patient, surgical debulking was performed with good clinical outcome. The Simpson grade of resection was grade II (complete tumour removal with coagulation of dural attachment), which is associated with a recurrence rate of approximately 10-15% over 10 years [17]. ABPA Management The concurrent diagnosis of ABPA in our patient required separate management. ABPA is characterized by hypersensitivity to Aspergillus antigens and typically presents with asthma, recurrent pulmonary infiltrates, and bronchiectasis. The diagnosis is based on clinical, radiological, and immunological criteria, according to the ISHAM working group criteria, which our patient fulfilled despite a lower total IgE level than typically seen [5]. Management of ABPA involves:
Monitoring: Regular assessment through total serum IgE levels, pulmonary function tests, chest imaging, and follow-up sputum cultures. Our patient's ABPA was managed with this comprehensive approach, with good control of pulmonary symptoms during the follow-up period. A reduction in total serum IgE levels from 365.5 IU/mL to 180 IU/mL over six months, along with the absence of A. terreus in follow-up sputum cultures indicated good response to therapy. Specific Considerations for A. terreus-induced ABPA The identification of A. terreus as the causative agent of ABPA in our patient has significant therapeutic implications:
Conclusion
LIMITATIONS
Author contribution:
We also declare that no-one who qualifies for authorship has been excluded from the list of authors. Ethical considerations: Free and informed consent for publication was obtained from the patient. Funding: None. Conflict of interest: None. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
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