A case of allergic fungal sinusitis caused by Bipolaris spicifera]

We describe a case of allergic fungal sinusitis (AFS) caused by Bipolaris spicifera, the first case reported in Japan. A 70-year-old man was admitted to our hospital because of diplopia following bilateral nasal obstruction and discharge. Radiological studies using computed tomographic scan showed a large soft tissue mass occupying the right frontal, bilateral ethmoid and sphenoid sinuses. He underwent drainage surgery and histopathological examination of the contents of the paranasal sinuses revealed scattered fungal hyphae within "allergic mucin". By cytological examination, these hyphae showed septation at irregular intervals, and were swollen to various sizes. Microbiological studies identified the fungus as Bipolaris spicifera. The presence of allergic mucin and scattered fungal hyphae were very important findings in making a diagnosis of AFS histopathologically, so squash cytology of the contents of the paranasal sinuses was quite useful to observe fungal elements and identify the strain in this case.     

Invasive fungal sinusitis and meningitis due to Arthrographis kalrae in a patient with AIDS

We report the first described case of Arthrographis kalrae pansinusitis and meningitis in a patient with AIDS. The patient was initially diagnosed with Arthrographis kalrae pansinusitis by endoscopic biopsy and culture. The patient was treated with itraconazole for approximately 5 months and then died secondary to Pneumocytis carinii pneumonia. Postmortem examination revealed invasive fungal sinusitis that involved the sphenoid sinus and that extended through the cribiform plate into the inferior surfaces of the bilateral frontal lobes. There was also an associated fungal meningitis and vasculitis with fungal thrombosis and multiple recent infarcts that involved the frontal lobes, right caudate nucleus, and putamen. Post mortem cultures were positive for A. kalrae.     

Les sinusites aspergillaires : à propos de trois cas dans l’hôpital de Sfax,Tunisie

Among the non invasive fungal sinus diseases, fungus ball has been increasingly reported and large published series have allowed better characterization of the disease and the treatment strategies in immunocompetent people. We report three cases of aspergillosis sinusitis diagnosed in our hospital. This fungal ball occurred in two men and one immunocompetent woman with a chronic sinusitis. Dental care by zinc oxide was found in one patient. The endoscopy and computed tomography have viewed training polypoides bridging the maxillary sinus only in the first case and the frontal sinus and ethmoid in the second case. A multi location has been found in the third case with achieving the ethmoid on both sides of the right maxillary sinus, right frontal sinus and sphenoidal sinus. The mycological and anatomopathological exam showed fungal hyphae but not invasion in all cases. Aspergillus flavus has been isolated in the third case. All the patients had only surgical treatment without antifungal. No recurrence was noted.DiscussionEndodontic treatment on maxillary teeth with zinc constitutes a strong risk factor for sinus fungus ball. However, the clinical symptoms may be expressive particularly in multisinus localisation, rarely reported. Functional endoscopic sinus surgery is the gold standard for treatment and antifungal therapy is unnecessary.     

Maxillary sinusitis caused by Pleurophomopsis lignicola.

An immunocompetent 59-year-old man developed sinusitis over a 6- to 8-month period after cutting down a rotted maple tree (Acer sp.). A polypoid obstruction with a bloody drainage was evident in his right nasal cavity. A computed tomographic scan showed an opacification of the maxillary sinus. Surgery was performed to remove a fungus ball that had extended into the patient's medial sinus cavity. Sections of the sinonasal mucosa revealed marked acute and chronic sinusitis with inflammation, congestion, and hemorrhage. Sections from the pasty brown to black debrided material revealed a fungus ball consisting of an extensive network of brown-pigmented, septate, profusely branched hyphae. When grown on oat agar, the phaeoid fungus produced pycnidia and was identified as Pleurophomopsis lignicola. The genus Pleurophomopsis includes seven species, which are all known from plant material. This report documents for the first time a coelomycetous fungus, P. lignicola, causing sinusitis in an immunocompetent patient.     

Esthesioneuroblastoma of the nose in a child

A rare case of nasal esthesioneuroblastoma in a 5-year-old child is described. The tumor involved the maxillary and ethmoid sinuses, destroyed superior compartments of nasal septum and medial wall of left orbit. In spite of the advanced process, there were hardly any clinical manifestations. Radical surgery with tumor excision and electroresection of the upper maxilla was effective. No tumor recurrence was found in a 2-year follow-up.     

Primary diffuse large B-cell lymphoma of the ethmoid sinus

Malignant lymphoma of the ethmoid sinus is very rare. A case of diffuse large B-cell lymphoma (DLBCL) of the left ethmoid sinus is presented here. A 79-year-old Japanese man was consulted to our hospital because of head ache and disturbance of left eye movement. Nasal endoscopy revealed a tumor, and imaging modalities including CT and MRI detected a tumor in the left ethmoid sinus. The tumor was invasive into left eye and left nose. A biopsy was performed via the nasal cavity. The biopsy revealed a diffuse proliferation of atypical lymphocytes. The atypical lymphocytes were large and had enlarged hyperchromatic nuclei. Mitotic figures were scattered. Hodgkin's cells were absent. Follicular structures were not seen. Immunohistochemically, the tumor cells were negative for cytokeratins (AE1/2, polyclonal, KL-1, and CAM5.2, Dako) and epithelial membrane antigen, CD3, CD15, CD30, CD45RO, and TdT. In contrast, the tumor cells were positive for CD20, CD45, CD79α, and p53. KI-67 labeling was 100%. Light chain restriction was present; there were numerous λ-chain-positive cells, while κ-chain-positive cells were scant. The pathological diagnosis was DLBCL of the left ethmoid sinus. Imaging of the whole body revealed no tumors and lymphadenopathy other than the ethmoid DLBCL. The patient was treated with chemoradiation, and is now alive 3 months after the presentation. In conclusion, a very rare case of DLBCL of the ethmoid sinus was reported.     

Bilateral ethmoid sinusitis with unilateral proptosis as an initial manifestation of metastatic prostate carcinoma.

This article presents a case of bilateral ethmoid sinusitis with unilateral proptosis as a presenting sign of an unsuspected prostate carcinoma. A 59-year-old Hispanic male presented to his primary care physician with nasal congestion and rhinitis. He was treated with antibiotics and antihistamine decongestants for 3 weeks without improvement. A trial of steroids resulted in brief improvement followed by a rapid onset of nasal obstruction with proptosis. A computed tomography scan revealed opacification of the ethmoid sinus with right proptosis. The presumptive diagnosis was orbital cellulitis secondary to chronic ethmoid sinusitis. Endoscopic sinusotomy and bilateral ethmoidectomies were performed. Biopsy results returned as metastatic adenocarcinoma, probably of prostate origin. Urological work-up and evaluation with biopsy confirmed the diagnosis of prostatic carcinoma. The patient was treated with chemotherapy and radiation therapy. He died 7 months later with disseminated disease.     

Fine‐needle aspiration biopsy as an initial diagnostic modality in a clinically unsuspected case of invasive maxillary fungal sinusitis: A case report

Cases of invasive fungal lesions involving the paranasal sinuses are generally diagnosed either on histologic examination or on fungal culture. Here, we report a case of invasive fungal sinusitis diagnosed primarily by a fine‐needle aspiration biopsy (FNAB). Our patient was a 36‐year‐old male who presented with a history of slowly progressive, painless swelling over the left cheek. The only positive clinical finding was a bony hard swelling in the left maxillary region, which was clinically interpreted as “fibrous dysplasia.” A computed tomographic (CT) examination of the paranasal sinuses showed a soft tissue attenuation lesion involving the bilateral maxillae, with the destruction of multiple bones and involvement of multiple sinuses and the bilateral orbits. FNAB of the maxillary swelling showed several giant cells, many of them exhibiting ingested organisms with a morphologic resemblance to the Aspergillus species of fungi. Strikingly, no significant inflammatory cells were seen on cytologic smears. Unfortunately, owing to a lack of initial clinical suspicion, as well as the patient's loss to further follow‐up, a culture/histopathologic examination could not be carried out. This case is presented chiefly to highlight the clinical utility of a simple FNAB procedure, as an initial diagnostic modality in cases of fungal sinusitis, which can masquerade clinically as a neoplastic lesion. In addition, if radiologic findings are also available at the time of cytologic examination, a pretherapeutic comment on the invasive nature of the lesion can also be made. Diagn. Cytopathol. 2010. © 2009 Wiley‐Liss, Inc.     

Orbitomaxillary mucormycosis (zygomycosis) and the surgical approach to treatment: perspectives from a maxillofacial surgeon

Rhinocerebral or rhino-orbitocerebral (mucormycosis) zygomycosis (ROCZ) usually occurs among patients with poorly controlled diabetes mellitus (especially those with ketoacidosis), solid malignancies, iron overload or extensive burns, in patients undergoing treatment with glucocorticosteroid agents, or in patients with neutropenia related to haematologic malignancies. The disease process starts with inhalation of the fungus into the paranasal sinuses. The fungus may spread to invade the palate, sphenoid sinus, cavernous sinus, orbits or cranially to invade the brain. Pain and swelling precede oral ulceration and the resulting tissue necrosis can result in palatal perforation. Infection can sometimes extend from the sinuses into the mouth and produce painful, necrotic ulcerations of the hard palate. If untreated, infection usually spreads from the ethmoid sinus to the orbit, resulting in the loss of extraocular muscle function and proptosis. Surgical treatment includes the resection of involved tissues of the face, including skin and muscle, any skin of the nose that is involved, maxillary and ethmoid sinuses, necrotic tissue of the temporal area and infratemporal fossa, and orbital exenteration. The keys to successful therapy include suspicion of the diagnosis and early recognition of the signs and symptoms, correction of underlying medical disorders such as ketoacidosis, and aggressive medical and surgical intervention.     

Longterm survival in acute rhinocerebral mucormycosis with giant cell arteritis and foreign body granulomas

A case of rhinocerebral mucormycosis occurring in a 41-year-old man with insulin-treated diabetes mellitus is reported. Microscopically, biopsy samples obtained from the left ethmoid and middle turbinate sinuses contained fungi that formed mycotic granulomas associated with multinucleate giant cell arteritis. The multinucleate giant cells contained broad, infrequently septate hyphase consistent with mucormycosis. The patient received surgical debridement with extenteration of the left orbit, and intravenous liposome-encapsulated amphotericin B. After 12 months, examination of the patient revealed complete healing. Multinucleate giant cell granulomas and arteritis are only exceptionally associated with rhinocerebral mucormycosis, but these histologic findings may be correlated with a progressive disease with better prognosis.     

鼻旁窦的年龄断面解剖学研究

目的：为影像学诊断和鼻旁窦手术的最佳年龄提供解剖学依据。方法：采用三组年龄的国人头部标本２９例，各组标本均按等比例分别切割为横切面、矢状切面和冠状切面。结果：各组标本均可见明显的上颌窦和筛窦；幼儿组未见显著的额窦和蝶窦。学龄组５０％未见明显的额窦和筛窦，余组标本可见额窦和蝶窦。成人上颌窦开口的长轴呈水平状，儿童上颌窦开口为向外下方的斜形管道。结论：鼻旁窦的形态有明显的年龄变化，窦额窦和蝶窦的最初气     

Botryomycosis ('bacterial ball') of the sinonasal tract caused by Pseudomonas aeruginosa

BACKGROUND: Botryomycosis is a chronic bacterial infection that typically presents as a cutaneous lesion. Visceral involvement may occur, but mucosal disease is uncommon. We report two cases of sinonasal tract botryomycosis that clinically simulated a neoplasm. METHODS: Two cases of sinonasal tract botryomycosis were identified from the Otolaryngic Tumor Registry at the Armed Forces Institute of Pathology, Washington, DC. The clinical records, slides, and paraffin blocks were available for both cases. Histochemical stains, including Brown and Hopps, Gomori's methenamine-silver, acid-fast bacilli, mucicarmine, periodic acid-Schiff, and Warthin-Starry, were performed. RESULTS: The patients were an 81-year-old man and a 43-year-old woman. The man presented with acute ethmoiditis and a bulging eye. Radiographic studies showed a soft tissue mass in his left maxillary antrum with osseous erosion of adjacent anatomic sites. The woman presented with persistent headaches of more than 1 year's duration, with increasing severity in the months prior to presentation. An expansile soft tissue mass was identified in her right maxillary and ethmoid sinuses. Surgery was performed on both patients. The histology included amorphous, acellular material and separate, rounded eosinophilic granules associated with a neutrophilic infiltrate. A Splendore-Hoeppli phenomenon was seen. Filamentous gram-negative bacilli, identifiable only by histochemical staining, were morphologically compatible with Pseudomonas aeruginosa. Cultures of samples taken from both patients intraoperatively confirmed the organisms as P. aeruginosa. CONCLUSIONS: Sinonasal botryomycosis is a rare localized disease that may be mistaken clinically for an aggressive neoplasm. Complete surgical evacuation is curative.     

Malignant giant cell tumor of the sphenoid

Malignant giant cell tumors (MGCTs) of the sphenoid sinus are extremely rare neoplasms. They are challenging to diagnose and difficult to treat because of their skull base location. To the best of our knowledge, we report the first case of a primary MGCT of the sphenoid arising in a patient with Paget's disease. A 77-year-old man presented with epistaxis and a history of Paget's disease. There was normal cranial nerve function although radiographic images disclosed a large mass centered in the sphenoid sinus and extending into the ethmoid and maxillary sinuses. Excisional biopsy revealed a MGCT composed of a cellular stroma with increased mitotic activity and necrosis with giant cells present throughout. Additional therapy was declined and the patient died with disease 7 months later. Because of their rarity, no treatment guidelines exist for the management of MGCTs of the sphenoid. We discuss both the diagnostic and therapeutic considerations based on a review of the pertinent literature.     

首诊眼科的真菌性鼻窦炎的影像解剖学特点

目的　分析以眼部症状为主的真菌性鼻窦炎的影像解剖学特征及临床表现。 方法　选取2008年1月~2018年1月三家医院耳鼻咽喉头颈外科收治的真菌性鼻窦炎患者285例中首诊于眼科的20例患者的临床资料,分析其特征及临床误诊的原因。 结果　该20例首诊于眼科的病例其病变主要位于筛窦3例,蝶窦2例,上颌窦合并筛窦3例,筛窦合并蝶窦8例,上颌窦合并筛窦、蝶窦4例,眼部症状主要为单侧眼胀痛、视力下降、视野改变、面部疼痛及头痛。 结论　以单侧眼部胀痛及视力改变就诊于眼科的患者,应考虑真菌性鼻窦炎的可能,及时给予鼻窦CT检查后转耳鼻咽喉科进行治疗。     

Nasal polyposis and fungal Schizophyllum commune infection: a case report

We present a rare case of eosinophilic fungal rhinosinusitis with nasal polyps in a 32-year-old woman caused by basidiomycete fungus Schizophyllum commune. Diagnosis was done by the endoscopic nasal examination, computed tomography (CT) of the paranasal sinuses, the histopathological examination of polyps, the presence of eosinophils and fungal hyphae in nasal mucus and by the detection of S. commune by culture. The patient was successfully treated by combination of oral itraconazole and topical corticosteroid therapy combined with surgery. The pathogenesis and diagnosis of allergic fungal rhinosinusitis are also discussed.     

A case of trichophytia profunda acuta of the glabrous skin]

We report the case of a 68-year-old man with eruption on his left arm in the lesion where he wore his wrist watch. He was treated with topical steroid ointment at another clinic. He also suffered from tinea pedis. Examination of the scale and hair showed hyphal elements. Histopathological examination revealed granulomatous reaction around the hair follicles. We found no fungal elements in the tissue in spite of detecting serial sections of them. Trichophyton rubrum was cultured from tissue and scale, and the case was diagnosed as Trichophytia profunda acuta of the glabrous skin. Spontaneous remission was shown only by topical treatment for tinea pedis. A therapy of "wait and see" may be one choice.     

Corneal ulcer due to Neocosmospora vasinfecta in an immunocompetent patient.

We report a case of Neocosmospora vasinfecta keratitis in a 55-year-old man. While the patient did not recall any specific trauma or eye injury, he might have sustained a trivial wound during the course of his duties as a farmer. Direct examination of corneal scrapings revealed fungus filaments. As topical treatment with natamycin and econazole and subsequent systemic ketoconazole therapy failed, a full thickness therapeutic keratoplasty was performed. Post-operative treatment with amphotericin B and clotrimazole combined with cyclosporine resulted in a complete cure. The residual corneal infiltration in the recipient cornea became clear in a week. The fungal isolate was initially identified as a Fusarium species, but later reidentified through the use of morphological characteristics and sequence analysis of the internal transcribed spacer region as N. vasinfecta. The latters is a Hypocrealean fungus not hitherto reported as a causative agent of keratomycosis.     

Metastatic esthesioneuroblastoma in a horse

A 17-year-old horse developed severe proptosis of the left eye over a period of 1.5 years. At post-mortem examination a neoplasm was found involving the left ethmoid bone, left maxillary sinus, left orbit, left superior turbinate, and the left eye. Tumour cells were arranged in nests separated by a fine fibrovascular stroma. Immunohistochemically, the tumour cells were labelled by antibodies against neurofilament protein, synaptophysin, glial fibrillary acidic protein and S-100 protein antigen, but were negative for chromogranin A, cytokeratin and desmin. Electronmicroscopically, the cells showed neurosecretory granules with an electron-dense centre and a light halo, and microfilaments. On the basis of macroscopical, light microscopical and ultrastructural findings a diagnosis of a metastatic esthesioneuroblastoma was made.     

Mycosis in the ear, nose and throat]

Recent trends of fungal infections of the ear, nose and throat were introduced from the viewpoint of otolaryngologic practice. Aspergillus terreus was the most common pathogen of otomycosis followed by A. niger and A. flavus. Lanoconazole showed the most effective antifungal function for these Aspergillus species by drug sensitivity test. Biological differences between clinical and soil-borne strains of A. terreus were evaluated. The clinical strains showed slower growth-rate on malt extract agar and different patterns of fingerprinting by Random Amplified Polymorphic DNA. Aspergillosis is the most common fungal disease in the paranasal sinuses. Unilateral opacity of the maxillary sinus which contains flecks of calcification was specifically found by CT-study. Surgical removal of the fungus ball and establishment of a drainage route to the nasal passage by endoscopic sinus surgery are effective to manage aspergillomas in paranasal sinuses. Although candidosis is a common and mild infection in the oral mucosa, underlying problems related to immunodeficiency syndrome must be evaluated.     

Myoepithelial carcinoma of the orbit: a clinicopathological and histopathological study

Tran TL, Broholm H, Daugaard S, Fugleholm K, Poulsgaard L, Prause JU, Kennedy SM, Heegaard S. Myoepithelial carcinoma of the orbit: a clinicopathological and histopathological study. APMIS 2010; 118: 324–30. Two cases of invasive myoepithelial carcinoma arising from the paranasal sinuses and invading the orbit are presented. Patient 1, a 53‐year‐old man, had a 3‐month history of proptosis, pain and epiphora of the right eye. The second patient, a 24‐year‐old man, had for a week been complaining of protrusion of his left eye and of orbital pain. Computed tomography scan and magnetic resonance imaging revealed tumour masses in the frontal, ethmoidal and maxillary sinuses with invasion of the orbit and the frontal lobe. Biopsies from both cases showed spindle and epithelioid tumour cells. Mitotic figures were frequent. Immunohistochemical staining showed positive reaction for bcl‐2, calponin, cytokeratins, CD99, S100, muscle‐specific antigen, smooth muscle antigen and vimentin. The Ki‐67 index was between 30–50% and 5–25%, respectively. Ultrastructurally, intermediate filaments, perinuclear tonofilaments and desmosomes were present. Based on these findings, a diagnosis of myoepithelial carcinoma of mixed cell type in both cases was evident. Both patients died shortly after the diagnosis was made even though both underwent radical surgery. Myoepithelial carcinoma of the paranasal sinuses is very rare and only six cases have been reported previously. We present the first two cases of myoepithelial carcinoma in the paranasal sinuses with invasion of the orbit. This is also the first report of myoepithelial carcinoma arising in the ethmoidal sinus.