Journal of Yeungnam Medical Science

Case report

Invasive sphenoid sinus aspergillosis with normal findings on initial diagnostic tests that mimics Tolosa-Hunt syndrome—a diagnostic dilemma: a case report: Yu Hun Jeong, Jongsuk Choi, Byung-Jo Kim, Hung Youl Seok; J Yeungnam Med Sci. 2023;40(2):198-201. Published online March 11, 2022; DOI: https://doi.org/10.12701/jyms.2021.01494

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Invasive sphenoid sinus aspergillosis can mimic Tolosa-Hunt syndrome (THS), leading to frequent misdiagnoses and potentially fatal consequences. We report a case of invasive sphenoid sinus aspergillosis initially misdiagnosed as THS. A 79-year-old man presented with right periorbital pain, ophthalmoplegia, and loss of vision. Initial evaluations including magnetic resonance imaging (MRI), were normal. He was first diagnosed with THS based on clinical features. The disease progressed despite high-dose intravenous steroid treatment, and an enhancing mass-like lesion was found in the right orbital apex, cavernous sinus, and sphenoid sinus on follow-up MRI. Aspergillosis was eventually confirmed by sphenoid sinus biopsy. The patient developed cerebral infarction and finally died despite being treated with amphotericin B. Given that invasive sphenoid sinus aspergillosis may initially resemble THS, high suspicion and rapid histological examination are important for diagnosis.

Citations

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Invasive Aspergillosis Mimicking Tolosa-Hunt Syndrome: A Case Report
Soo-Im Jang, Seong Yeon Park, Sungim Choi, Eun-Ja Lee, Soo-Hyun Park, Nam-Hee Kim
Korean Journal of Neuromuscular Disorders.2023; 15(2): 46. CrossRef
Challenging Diagnosis of Invasive Sinus Aspergillosis Mimicking Gradenigo’s Syndrome in an Elderly Patient with T-Cell Lymphoma
Victoria Ramos de Ascanio, Gloria Liaño-Esteso, David Roldán, Teresa Collazo-Lorduy, Sara Martínez-Flores, José Ángel Hernández-Rivas, Isabel González-Gascón-y-Marín
Geriatrics.2023; 9(1): 4. CrossRef

Case Reports

Adrenocorticotropic hormone (ACTH)-producing pheochromocytoma presented as Cushing syndrome and complicated by invasive aspergillosis.: Jae Ho Cho, Da Eun Jeong, Jae Young Lee, Jong Geol Jang, Jun Sung Moon, Mi Jin Kim, Ji Sung Yoon, Kyu Chang Won, Hyoung Woo Lee; Yeungnam Univ J Med. 2015;32(2):132-137. Published online December 31, 2015; DOI: https://doi.org/10.12701/yujm.2015.32.2.132

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Abstract PDF

Adrenocorticotropic hormone (ACTH)-producing pheochromocytoma has been rarely reported, whereas only a few cases of Cushing syndrome accompanied by opportunistic infections have been reported. We experienced a patient with pheochromocytoma with ectopic Cushing syndrome complicated by invasive aspergillosis. A 35-year-old woman presented with typical Cushingoid features. Her basal plasma cortisol, ACTH, and 24-hour urine free cortisol levels were significantly high, and 24-hour urine metanephrine and catecholamine levels were slightly elevated. The endogeneous cortisol secretion was not suppressed by either low- or high-dose dexamethasone. Abdominal computed tomography (CT) revealed a heterogeneous enhancing mass measuring approximately 2.5 cm in size in the left adrenal gland. No definitive mass lesion was observed on sellar magnetic resonance imaging. On fluorine-18 fluorodeoxyglucose positron emission tomography/CT, a hypermetabolic nodule was observed in the left upper lung. Thus, we performed a percutaneous needle biopsy, which revealed inflammation, not malignancy. Thereafter, we performed a laparoscopic left adrenalectomy, and its pathologic finding was a pheochromocytoma with positive immunohistostaining for ACTH. After surgery, the biochemistry was normalized, but the clinical course was fatal despite intensive care because of the invasive aspergillosis that included the lungs, retina, and central nervous system.

Citations

Citations to this article as recorded by

Ectopic ACTH- and/or CRH-Producing Pheochromocytomas
Patrick F Elliott, Thomas Berhane, Oskar Ragnarsson, Henrik Falhammar
The Journal of Clinical Endocrinology & Metabolism.2021; 106(2): 598. CrossRef
Ectopic adrenocorticotrophic hormone syndrome (EAS) with phaeochromocytoma: a challenging endocrine case with a happy ending
Sharifah Faradila Wan Muhamad Hatta, Leoni Lekkakou, Ananth Viswananth, Harit Buch
BMJ Case Reports.2019; 12(8): e230636. CrossRef
Severe Cushing Syndrome Due to an ACTH-Producing Pheochromocytoma: A Case Presentation and Review of the Literature
Jenan N Gabi, Maali M Milhem, Yara E Tovar, Emhemmid S Karem, Alaa Y Gabi, Rodhan A Khthir
Journal of the Endocrine Society.2018; 2(7): 621. CrossRef

A case of Bilateral Near Blindness Secondary to Isolated Sphenoid Sinus Aspergillosis with Headache.: Jun Pil Yoon, Se Jin Lee, Jun Lee, Ju Hyun Kim, Hyun Doo Noh; Yeungnam Univ J Med. 2007;24(1):79-84. Published online June 30, 2007; DOI: https://doi.org/10.12701/yujm.2007.24.1.79

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Abstract PDF: Sphenoid sinus aspergillosis is notorious for its serious complications, such as permanent cranial nerve deficits and possible death. The most common associated symptoms are headache, followed by visual changes, and cranial nerve palsies. Because of an insidious onset, frequently resulting in missed and delayed diagnosis, sphenoid sinus aspergillosis is a potentially lethal medical condition. We report a case of visual loss secondary to isolated sphenoid sinus aspergillosis. A 69-year-old man presented to our hospital with the complaint of headache. The headache started one year previously and was described as severe dull pain localized bilaterally to the temporo-orbital region. The patient took daily NSAIDs for the pain. The neurological examination was normal. The MRI of the brain showed a left sphenoid sinusitis. A transnasal endoscopic superior meatal sphenoidotomy was performed. Aspergillosis was confirmed after a surgical biopsy was obtained. The patient was discharged from hospital without antifungal therapy. One month later, the patient complained of headache and loss of vision bilaterally. The orbital MRI showed a left cavernous sinus and bilateral optic nerve invasion. The loss of visions was permanent. In our case, the diagnosis was delayed; antifungal agents were not administered after surgery and the patient lost his vision as a result. Therefore, early diagnosis and proper treatment are important. Although the treatment of an invasive type of aspergillus has not been established, surgical removal of a nidus and aggressive antifungal therapy are recommended.

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