History Patient, male, 36 years old. He was diagnosed with nasal congestion for 10 months, redness and swelling on the back of the nose for 7 months, and itching on September 19, 2005. 10 months ago, he felt nasal congestion and dizziness without any obvious cause, and underwent a coronal CT of sinuses in a hospital in Zhejiang Province, which showed hypertrophy of the right inferior turbinate and deviation of the nasal septum, and underwent surgery for deviation of the nasal septum. Seven months ago, a “red spot” appeared on the skin of the right nasal flank, which itched and gradually worsened nasal congestion. A review of cranial CT at a local hospital showed “inflammation of the right maxillary sinus, septal sinus and nasal cavity, and curvature of the nasal septum”. After that, the lesions gradually increased to the forehead, the whole nose, cheeks and upper lip and became lumpy, itchy, unable to sleep and only breathing through the mouth. Afterwards, he visited several hospitals and underwent several tests such as nasal endoscopic biopsy, skin histopathology and fungal culture, but no clear diagnosis was made. On September 19, 2005, he was admitted to our outpatient clinic. He had no fever during his illness. He denied any previous history of other chronic diseases such as diabetes and trauma. The patient grew up in Yihuang County, Jiangxi Province, and was a farmer, growing only rice at home. As an adult, he worked in Taizhou, Zhejiang Province for a long time and was engaged in shoe tread work. He was married and both mother and son were healthy. No special medical history in the family.
Physical examination: body temperature 36.8℃, pulse 78, blood pressure 120/80mmHg, respiration 20 times/min. Odd facial features. Mental health is fair. There was no significant enlargement of superficial lymph nodes behind the ear, under the jaw and neck. No heart or lung abnormalities were observed. Otorhinolaryngology The soft tissue in the upper part of the right nasal vestibule was swollen, thickened and connected to the inferior turbinate, and the mucosa was still smooth. The structure of the sulcus of the middle turbinate of the nose was normal. The lesion of the left nasal vestibule was similar to that of the right but less severe. Dermatological condition The middle of the forehead was about 1/3 wide to the whole nose, inner cheek, and upper lip were severely diffusely swollen and deformed, and the fossa at the root of the nose disappeared. The surface was smooth, dark red, with still clear margins, and the pressure pain was not obvious and hard.
Biochemical examination Blood routine: normal range; urine routine: normal range; sedimentation: 21 mm/1h Anti-O: 87 IU/mL. immunological indexes: cellular immunity CD3 63.59%; CD4 31.39%; CD8 24.03%; CD4/CD8 1.31%.
Humoral immunity IgG 2290.00 mg/dl (751.00-1560.00) IgA 155.00 mg/dl (82.00-453.00) IgM 318.00 mg/dl (46.00-304.00) C3 125.00 mg/dl (79-152) C4 53.70 mg/dl (16.00 -38.00).
Nasal imaging (the patient brought the results from outside hospital) 1. X-ray: normal nasal bone structure, no signs of bone destruction, soft tissue swelling in the nose. 2. CT findings: mucosal hypertrophy of the right maxillary sinus, sieve sinus, and middle and upper nasal tract, sinus wall bone structure is still intact. The diagnosis was inflammation of the right maxillary sinus, sieve sinus and nasal cavity. 3. MRI images showed that the nasal wing root and surrounding local structures were seen as T1 and slightly long T2 lesions, the nasal wing was thickened, the lesion was not clearly demarcated from the normal tissue structure, the right turbinate was hypertrophied, the right maxillary sinus cavity was seen as T1 and long T2 signal shadow, and the bone was not clearly destroyed. The initial diagnosis was: ① abnormal signal in the nasal root and surrounding local structures, hypertrophy of the right turbinate. (infectious granuloma) ② right maxillary sinusitis.
Histopathological examination Histopathological examination showed granulomatous changes, with small focal necrosis in some granulomas, and a large number of lymphocytes infiltrating around the histocytes, while eosinophilic leukocytes were scattered infiltrating in the granulomas. (Splendore-Hoeppli phenomenon).
Nasal fungal examination 1, direct microscopic examination: after local anesthesia, several pieces of brittle cartilage-like tissue were removed from the right nasal cavity under the nasal endoscope, and the tissue was shredded with sterile ophthalmic scissors and filmed with 10% potassium hydroxide. 2, fungal culture: the shredded tissue was inoculated in sandberg agar medium (SDA), potato medium (PDA), and brain heart juice medium (BHI). . Multiple tissue cultures were of the same strain. Colony characteristics: vigorous growth at room temperature on SDA and PDA, 3 days colonies grew all over the slope. Yellowish white. Initially waxy texture, later the center surface is powder, the back is cerebral gyrus, the surrounding formation of satellite colonies (PDA more obvious). the growth rate of colonies on SDA is faster than the growth rate of PDA. the growth rate of colonies on BHI is basically the same as PDA, but the morphology of colonies and SDA the same. Microscopic features: mycelium is wide, no separation. Mycelium apical partition into sporangiophore, then grow primary sporangiam in tadpole shape. The sporangiophore is round or pear-shaped, and has a conical papilla with a rounded top, similar to a light bulb. Some of the mycelium is dilated at both ends or forms a dumbbell shape with secondary conidia. Some sporocysts budding, producing many radially distributed secondary microconidia, thus forming a sporocyst corona. Villous spores (villose conidia) with villous growth can also be seen. Usually after the 8th-9th day, spore production is significantly reduced, while a large number of thick non-septate mycelium can be seen in the periphery of the colony.
Clinical diagnosis: otomycosis Treatment: 10 ml of 10% potassium iodide (KI) orally 3 times a day and 1.0 g of synthroid tablets twice a day. 7 d later KI was increased to 20 ml/dose, and then gradually increased to 30 ml/dose after tolerance. After half a month of follow-up, the itching was significantly reduced to none, and the lesions were smaller and softer in texture than before, and the nasal root fossa was obvious. Later, drug resistance was developed, and it was changed to spironolactone (itraconazole) and lamisil (terbinafine) for one and a half years, and finally cured.
Discussion Entomophthoramycosis, also known as subcutaneous algae, includes two groups of diseases, basidiobolomycosis and conidiobolomycosis. They are histologically similar, but differ in clinical and fungal manifestations. Coronal otomycosis and heterosporous otomycosis are the two known pathogenic species causing otomycosis, with coronal otomycosis accounting for about 90%. The clinical manifestation is a limited non-invasive disfiguring mass on the face of the nose, but in immunosuppressed individuals, it can cause a lethal disseminated infection. There are also reports of 2 cases of periorbital cellulitis in infants caused by Isospora otitis. The disease has typical clinical features: ① adult male, with a history of exposure to rice and a history of digging the nose by hand. ② Unilateral nasal congestion and enlargement of the inferior turbinate as the first symptom, later the skin tissue is red, swollen, hard and adheres closely to the tissue below, gradually involving the forehead, the whole nose, the cheek and the upper lip in a continuous mass, causing a strange appearance of the face. ③ There is no obvious pain in the mass, no systemic fever and other symptoms, and no obvious abnormalities in immune function. ④ The surrounding bone was not involved, the damage was clearly defined, and the skin surface was structurally intact. ⑤ Histopathology showed eosinophilic granuloma, and fungal cyst-like structures with positive PAS staining were seen in multinucleated giant cells. ⑥ Fungal culture can be seen on the flat dish lid ejected by the colony ejected spores formed by the same reflection as the colony shape. The colony microscopically see spherical sporangia, cone-shaped top round papillae obvious, also see more small sporangia crown. (7) It is effective for potassium iodide and sulfonamide treatment. Successful treatment with potassium iodide, ketoconazole, itraconazole, fluconazole, terbinafine, and amphotericin B alone as well as the combination of itraconazole and fluconazole, and terbinafine and amphotericin B have been reported abroad.