A 32-year-old male patient presented to the otolaryngology outpatient department after experiencing visible progressive facial misalignment for 3 weeks. He complained about the sunken appearance of his left eye and the asymmetric protrusion of his bilateral maxillary bone. However, he had no history of recent facial trauma, sinus surgery, or any nasal or ophthalmological symptoms. Physical examination revealed a deepening of the left superior orbital sulcus and left hypoglobus (Figure 1A). The patient's visual acuity and extraocular movements were normal. Nasal endoscopy indicated the absence of polyps, purulent secretion, edematous mucosa, or any other abnormality. Subsequent computed tomography (CT) revealed that all the walls of the left maxillary sinus were bowing inward, causing a considerable reduction in the sinus volume and a downward displacement of the orbital floor (Figure 1B,C). A diagnosis of silent sinus syndrome (SSS) was made. During endoscopic maxillary antrostomy performed under general anesthesia, total occlusion of the maxillary sinus ostium and the accumulation of mucus were discovered. Patency of the maxillary sinus was subsequently achieved. Six months after the maxillary antrostomy, the patient self-reported improved facial asymmetry (Figure 1D), and a follow-up CT scan revealed a substantial expansion of the size of the patient's maxillary sinus and an improvement in his enophthalmos (Figure 1E,F). The criteria for diagnosing SSS include spontaneous enophthalmos and/or hypoglobus, contraction of the maxillary sinus with osteomeatal complex (OMC) occlusion on CT images as well as the absence of symptoms of sinonasal inflammatory disease, orbital trauma, or any congenital deformity.1-3 The pathophysiology of SSS involves maxillary sinus hypoventilation after total OMC occlusion. Subsequent gas resorption leads to sub-atmospheric pressure, creating a vacuum that causes sinus wall retraction. Over time, the maxilla bone undergoes secondary demineralization, and the resulting negative sinus pressure causes the weakened areas to implode.4 This phenomenon was confirmed by Kass et al.5 a series of human studies. When SSS was first described in 1990s, the Caldwell-Luc procedure was the conventional surgical solution. However, the treatment trend has shifted to endoscopic surgery, which provides advantages such as reduced infection rates, a reduced risk of numbness, and improved maxillary sinus aeration. Nevertheless, the endoscopic procedure must be performed carefully to prevent orbital content from prolapsing into the maxillary sinus. A major debate is whether orbital floor reconstruction should be performed during maxillary surgery. Some surgeons prefer a one-stage approach, whereas others recommend a watch-and-wait strategy because spontaneous re-expansion and atelectasis resolution can occur to some extent after the maxillary ostium is widened.2-4 Our patient only underwent maxillary surgery, and at 6 months after the procedure, we observed the regression of enophthalmos and an increase in the maxillary sinus size. This result suggests that simultaneous endoscopic antrostomy and orbital floor reconstruction are not required. In summary, this report presents a rare clinical case of a patient who visited our institution solely because of aesthetic concerns. Furthermore, the patient was pleased with the cosmetic results achieved through accurate diagnosis and treatment. All authors declare no conflict of interest. Approval for retrospective data collection/secondary use of health information was obtained from the Institutional Review Board of Kaohsiung Medical University Hospital (KMUHIRB-E(I)-20230135).
Total thyroidectomy (TT) in patients with Graves’ disease is challenging even for an experienced thyroid surgeon. This study aimed to investigate the accumulation of experience and applying newly developed devices on major complications and voice outcomes after surgery of a single surgeon over 30 years. This study retrospectively reviewed 90 patients with Graves’ disease who received TT. Forty-six patients received surgery during 1990–1999 (Group A), and 44 patients received surgery during 2010–2019 (Group B). Major complications rates were compared between Group A/B, and objective voice parameters were compared between the usage of energy-based devices (EBDs) within Group B. Compared to Group B, Group A patients had higher rates of recurrent laryngeal nerve palsy (13.0%/1.1%, p = 0.001), postoperative hypocalcemia (47.8%/18.2%, p = 0.002), and postoperative hematoma (10.9%/2.3%, p = 0.108). Additionally, Group A had one permanent vocal cord palsy, four permanent hypocalcemia, and one thyroid storm, whereas none of Group B had these complications. Group B patients with EBDs had a significantly better pitch range (p = 0.015) and jitter (p = 0.035) than those without EBDs. To reduce the major complications rate, inexperienced thyroid surgeons should remain vigilant when performing TT for Graves’ disease. Updates on surgical concepts and the effective use of operative adjuncts are necessary to improve patient safety and voice outcome.
Aneurysmal bone cysts (ABCs) mostly occur in the metaphysis of long bones. Primary paranasal ABCs are extremely rare, and most reported cases reveal typical histopathological features including cystic space with fibrous septa and hemorrhage. Solid-variant ABCs or solid ABCs lacking cyst formation may be histologically indistinguishable from giant cell reparative granulomas, giant cell tumor of bone, and brown tumor. Here we report the case of a 24-year-old woman with a paranasal mass diagnosed as USP6-rearranged solid ABC, mimicking giant cell reparative granuloma, giant cell tumor of bone, and brown tumor. For paranasal sinus bone or soft tissue tumors containing numerous giant cells, molecular analysis including the USP6 gene may serve as a useful diagnostic tool to distinguish solid ABCs from other giant cell–rich lesions.
Few studies have investigated the relationship between chronic rhinosinusitis (CRS) and erectile dysfunction (ED). This case-control study aimed to investigate the association between CRS and the risk of ED in a large national sample. Tapping Taiwan’s National Health Insurance Research Database, we identified people 30 years or older with a new primary diagnosis of CRS between 1996 and 2007. The cases were compared with sex- and age-matched controls. We identified 14 039 cases and recruited 140 387 matched controls. Both groups were followed up in the same database until the end of 2007 for instances of ED. Of those with CRS, 294 (2.1%) developed ED during a mean (SD) follow-up of 3.20 (2.33) years, while 1 661 (1.2%) of the matched controls developed ED, mean follow up 2.97 (2.39) years. Cox regression analyses were performed adjusting for sex, age, insurance premium, residence, hypertension, hyperlipidemia, diabetes, obesity, coronary heart disease, chronic kidney disease, chronic obstructive pulmonary disease, asthma, allergic rhinitis, arrhythmia, ischemic stroke, intracerebral hemorrhage, and medications. CRS was revealed to be an independent predictor of ED in the fully adjusted model (HR = 1.51; 95% CI = 1.33–1.73; P < 0.0001).
Eosinophils are important inflammatory cells involved in the pathogenesis of chronic rhinosinusitis with nasal polyps (CRSwNP). Vitamin D and its derivatives, in addition to their classic role as regulators of electrolytes homeostasis, have modulatory effects in immunological and inflammatory responses. Such properties suggest that vitamin D might also play a role in inflammatory airway diseases such as CRSwNP. In this study, we investigated the effect of vitamin D derivatives (calcitriol and tacalcitol) on the secretion of eotaxin and Regulated on Activation, Normal T Cell Expressed and Secreted (RANTES), the two major eosinophil chemoattractants, in fibroblasts derived from the polyps of Taiwanese CRSwNP patients. Patients diagnosed with eosinophilic CRSwNP but without malignancies or asthma and undergoing elective endoscopic sinus surgery were recruited. Three primary fibroblast cultures were established using the polyp specimens obtained from these patients. The third to eighth passages of the fibroblasts were used for in vitro studies. Nasal polyp-derived fibroblasts were stimulated with IL-1β (10 ng/mL) for 24 hours, followed by replacement with media alone or with calcitriol or tacalcitol (10 μM) and incubation for another 24 hours. After the treatments, the levels of secreted eotaxin and RANTES were evaluated by ELISA assays. The results showed that IL-1β could substantially stimulate the secretion of eotaxin (p < 0.01) and RANTES (p < 0.01) in nasal polyp-derived fibroblasts. More importantly, this stimulatory effect was significantly suppressed by adding calcitriol (p ≤ 0.002 for eotaxin and p ≤ 0.008 for RANTES) or tacalcitol (p ≤ 0.009 for eotaxin and p ≤ 0.02 for RANTES). Therefore, the inhibitory effect of vitamin D derivatives on eotaxin and RANTES secretion might shed light not only on the disease mechanism, but also on the potential use of vitamin D in pharmacotherapy of Taiwanese patients with CRSwNP.
Vitamin D and its derivatives have modulatory effects in immunological and inflammatory responses. Such properties suggest that they might have an impact on chronic inflammatory airway diseases, including nasal polyposis. The aim of this study was to understand the role of vitamin D in chronic rhinosinusitis with nasal polyps (CRSwNP) by investigating its effect on the secretion of matrix metalloproteinase-2 (MMP-2) and MMP-9 in nasal polyp-derived fibroblasts. Two primary fibroblast cultures were established from nasal polyp tissues obtained during surgery. The nasal polyp-derived fibroblasts were stimulated with tumor necrosis factor-α (TNF-α; 10 ng/mL) for 24 hours, followed by replacement with media alone or with vitamin D derivatives (calcitriol or tacalcitol; 10μM) and incubated for another 24 hours. After the treatments, the levels of MMP-2 and MMP-9 secreted were evaluated by both enzyme-linked immunosorbent assay (ELISA) and Western blot analysis. ELISA results revealed that TNF-α could substantially stimulate the secretion of MMP-2 (p < 0.01) and MMP-9 (p < 0.001) in nasal polyp-derived fibroblasts. More importantly, such stimulatory effect was significantly suppressed by adding calcitriol (p ≤ 0.01 for MMP-2 and p < 0.001 for MMP-9) or tacalcitol (p < 0.005 for both MMP-2 and MMP-9). The ELISA results were also confirmed by Western blot analysis. The inhibitory effect of vitamin D derivatives on MMP-2 and MMP-9 secretion could potentiate their application in pharmacotherapy of Taiwanese CRSwNP patients.
Ocular chemical burn is a severe injury with poor outcomes. Immediate and appropriate management is highly related to prognosis. We studied the effect of cultured human adipose tissue-derived stem cells on the regeneration of the rabbit cornea after alkaline chemical burn, using used human adipose tissue-derived stem cells as the source material. Immediately after the chemical burn, the experimental eye received a single subconjunctival injection of a stem cell suspension (1.3 × 105 cells/0.2 mL), with the other eye serving as control. Rabbits were sacrificed and specimens taken 30 days after injection. The experimental group showed faster wound healing than the control group, and the result for the experimental group was clearer cornea medium. Histologically, there were five to six epithelial cell layers on the corneas of the experimental group as compared to two to three cell layers on the corneas of the control group. Wilcoxon signed rank test showed a significant difference in the epithelial cell layers between the two groups. Surface markers for connexin 43 (Cx43), β-catenin, E-cadherin, and P63 were analyzed. Cx43 and β-catenin showed significant change, as determined by the Wilcoxon signed rank test, which indicated good cell renewal during repair of the corneal epithelium damaged by the chemical burn. E-cadherin and P63 showed no significant change during the epithelium healing process. Transplantation of cultured human adipose tissue-derived stem cells as a treatment for a corneal chemical burn promotes cell renewal and assists in damage repair.
Background Vitamin D deficiency is reported to be associated with increased incidence of allergic airway diseases and is correlated with the severity of asthma. This study was designed to determine if serum Vitamin D level is lower in chronic rhinosinusitis with nasal polyposis (CRSwNP) patients and if low serum Vitamin D level is correlated with the severity of CRSwNP. Methods New CRSwNP patients undergoing elective endoscopic sinus surgery were recruited. Patients with malignancies or asthma were excluded. Twenty chronic rhinosinusitis without nasal polyposis (CRSsNP) patients were used as control. Demographic characteristic information was collected. The severity of CRSwNP was assessed with the Lund-Mackay (LM) score and polyp grading system. Vitamin D status was assessed by measuring circulating 25-hydroxyvitamin D (25OHD) by using commercial chemiluminescence immunoassay. Data were stratified by factors known to affect serum 25OHD, including sex, race, and body mass index. Results Serum 25OHD levels (ng/mL ± SD) were significantly lower in patients with CRSwNP (21.4 ± 5.7) than in those with CRSsNP (28.8 ± 6.2; p < 0.001). The incidences of vitamin D deficiency (<20 ng/mL) in CRSwNP and CRSsNP patients were 45.5 and 6.3%; however, the incidences of vitamin D insufficiency (20–30 ng/mL) in these patients were 50.0 and 62.5%, respectively. A significantly negative relationship was found between serum 25OHD level and polyp grade (r = −0.63; p = 0.001), indicating lower serum 25OHD was associated with higher polyp grade. Serum 25OHD was inversely related to both LM score and total IgE level as well; however, statistical significance was not found. Conclusion A significantly lower vitamin D level was found in a group of Taiwanese CRSwNP patients, which revealed an association with greater nasal polyp size. Serum vitamin D levels could be added to the routine workup of patients suffering from CRS and these data could be used to potentially help determine the disease severity.
Matrix metalloproteinase (MMP) is involved in the upper airway remodeling process. We hypothesized that MMP had an additive effect on the formation of recurrent nasal polyp. We also investigated the association between the functional promoter polymorphism of MMPs and the intensity of labeling index. Expressions of MMP-2 and MMP-9 were assessed via immunohistochemical staining and compared between different groups, including recurrent nasal polyps, nonrecurrent nasal polyps, and control nasal mucosa. Two promoter functional single-nucleotide polymorphisms (rs3918242 for MMP-9 and rs243865 for MMP-2) were selected to correlate with staining intensity. Expression of MMP-9 was significantly enhanced in gland for recurrent nasal polyp (p = 0.016) and nonrecurrent nasal polyp (p = 0.005) compared to the control. MMP-2 positivity was significantly increased in surface epithelium for recurrent nasal polyp (p = 0.004) compared to the control (p = 0.061). However, there was no significant difference in MMP-9 and MMP-2 expressions between recurrent and nonrecurrent nasal polyps. Genetic polymorphism of MMP-2 and MMP-9 functional promoters was not associated with the intensity of labeling index. These results suggested that up-regulation of MMP-9 in gland and MMP-2 in surface epithelium was characteristic of both recurrent and nonrecurrent nasal polyps. Pathogenesis of recurrent nasal polyps may involve a mechanism other than MMP.
Here is a rare case of lingual leiomyomatous hamartoma (LLH) with bifid tongue tip and tongue-tie in a patient with non-oral-facial-digital syndrome (OFDS). A 29-year-old male consulted for a painless tumor over the midline of the tongue dorsum measuring 2 × 1.5 cm. The tumor was excised and the tongue-tie was corrected. Diagnosis of LLH was based on histo-pathologic and immuno-histochemical studies. The epidemiologic data and differential diagnosis of LLH, as well as related literature, are discussed. To date, only 14 cases of LLH have been reported in English literature. This may be the first reported case of LLH with bifid tip and ankyloglossia in a non-OFDS patient.
BACKGROUND:Matrix metalloproteinase (MMP) is involved in the remodeling process of inflammatory airway diseases and is correlated with the severity of asthma. We hypothesized that MMP was associated with the severity of chronic rhinosinusitis with nasal polyps (CRSwNPs). We also investigated the effect of allergy on the expression of MMP in the polyp.METHODS:The expression of MMP-2 and -9 was investigated in recurrent nasal polyps of 30 patients and in nonrecurrent nasal polyps of 31 patients undergoing endoscopic sinus surgery. These expressions were then compared with those in control nasal mucosal samples obtained from 32 patients with chronic hypertrophic rhinitis. Demographic data, Lund-Mackay (LM) score, polyp grade, and allergy status were obtained for all patients. Tissue samples were assessed via immunohistochemistry.RESULTS:MMP-2 and -9 were constantly expressed in recurrent NPs, primary NPs, and control nasal mucosa. The expression of MMP-9 was significantly enhanced in glands and MMP-2 positivity was significantly increased in surface epithelium for patients with NPs when compared with control nasal mucosa. The expression of MMP-9 and -2 was not correlated with polyp grade and LM score. Allergic status is an independent factor in the expression of MMP-2 and -9.CONCLUSION:These results suggested up-regulation of MMP-9, and MMP-2 in gland and surface epithelium, respectively, were characteristic of NPs. Therefore, patients with allergy will exhibit greater MMP-2 and -9 positivity. However, the MMP-2 and -9 expression intensity was not correlated with the severity of CRS with nasal polyposis.
The study's purposes are to identify patient characteristics, treatment response and survival rate, and to describe the important prognostic factors for our patients with extranodal head and neck lymphoma. Furthermore, no study has systemically discussed the overall figure of this disease in Taiwan and we analyzed our data on this topic. A retrospective review was performed for 86 patients with extranodal head and neck lymphoma, diagnosed in Kaohsiung Medical University Hospital, between 1990 and 2007. We evaluated the medical records and analyzed the possible factors affecting treatment outcomes, survival rate, and free-from-disease (FFD) survival rate. Forty-nine male and 37 female patients were included with a male:female ratio of 1.32:1. The most frequent histologic type was diffuse large B cell lymphoma, accounting for 41.9% of the total. The most common primary site involved with extranodal head and neck non-Hodgkin's lymphoma was a tonsil with 27 cases (31.4%). Stage, international prognostic index (IPI) score, B symptoms, lactate dehydrogenase (LDH) level, and lymph node status significantly affected treatment response. The overall 5- and 10-year survival rates were 68.0% and 57.8%, respectively. The FFD survival rate was 53.6% and 49.3% at 5 and 10 years, respectively. Factors including stage, lymph node status, LDH level, and IPI score produced significant differences in both overall survival and FFD survival. Our analyzed information is similar to other previously presented studies. Stage, IPI score, B symptoms, LDH level, and neck nodal status can be used to evaluate the treatment outcomes. Neck nodal status and stage are the two significant prognostic factors for overall survival.
ObjectiveMatrix metalloproteinase (MMP) is involved in the upper airway remodeling process. We hypothesized that MMP had an additive effect to the formation of recurrent nasal polyp. We also investigated the association of MMP’s functional promoter polymorphism and its labeling index in different tissues.MethodThirty‐two patients with recurrent nasal polyps and 30 patients without recurrence of nasal polyp (non‐recurrent) after surgery were collected. Inferior turbinate mucosa obtained from 31 patients without chronic rhinosinusitis were used as controls. Two promoter functional single nucleotide polymorphisms were selected. Tissue samples were frozen for immunohistohemistry.ResultsThe expression of MMP‐9 was significantly enhanced in gland, but not in surface epithelium, for recurrent nasal polyp (P =. 016) and non‐recurrent nasal polyp (P =. 005) compared with the control. MMP‐2 positivity was significantly increased in surface epithelium, but not in gland, for recurrent nasal polyp (P =. 004), and a trend toward significance for non‐recurrent nasal polyp compared to the control. However, there was no statistically significant difference of MMP‐9 and ‐2 expressions between recurrent nasal polyp and non‐recurrent nasal polyp. The genetic polymorphism of MMP‐2 and ‐9 functional promoters was not associated with the intensity of immunohistochemical labeling index.ConclusionThese results suggested up‐regulation of MMP‐9 in gland and MMP‐2 in surface epithelium were characteristic of both recurrent nasal polyp and non‐recurrent nasal polyp. The pathogenesis of recurrent nasal polyps may involve a mechanism other than MMP.
OBJECTIVES:To review our experience with deep neck infections and identify the risk factors for developing complications and prolonged hospitalization.SUBJECTS AND METHODS:We analyzed the prospectively collected database of 439 patients with deep neck infections between January 1996 and July 2007 at a single institution. Their demographic data, duration of hospitalization, etiology, underlying systemic disease, bacteriologic and radiologic studies, complications, and treatment outcome were reviewed and analyzed. Patients with superficial abscess or cellulitis, necrotizing fasciitis, and peritonsillar abscess were excluded.RESULTS:Dental infection and upper airway infection remained the two most common etiologies. Coexisting head and neck malignancy was found in eight cases. Patients with systemic diseases and a C-reactive protein value of more than 100 microg/mL tend to develop complications, resulting in prolonged hospitalization.CONCLUSION:We should pay more attention to those patients with systemic diseases or C-reactive protein values more than 100 microg/mL. The current study represents the largest and longest analysis of deep neck infection in the available literature.
BACKGROUND:Matrix metalloproteinase (MMP) is involved in the upper airway remodeling process. We hypothesized that genetic variants of the MMP-9 gene are associated with cases of chronic rhinosinusitis with nasal polyposis.METHODS:We conducted a case-control study where 203 cases of chronic rhinosinusitis with nasal polyposis and 730 controls were enrolled. Three tagging single nucleotide polymorphisms (SNPs) and one promoter functional SNP rs3918242 were selected. Hardy-Weinberg equilibrium (HWE) was tested for each SNP, and genetic effects were evaluated according to three inheritance modes. Haplotype analysis was also performed. Permutation was used to adjust for multiple testing.RESULTS:All four SNPs were in HWE. The T allele of promoter SNP rs3918242 was associated with chronic rhinosinusitis with nasal polyposis under the dominant (nominal p = 0.023, empirical p = 0.022, OR = 1.62) and additive models (nominal p= 0.012, empirical p = 0.011, OR = 1.60). The A allele of rs2274756 has a nominal p value of 0.034 under the dominant model and 0.020 under the additive model. Haplotype analysis including the four SNPs showed a global p value of 0.015 and the most significant haplotype had a p value of 0.0045. We did not see any SNP that was more significant in the recurrent cases.CONCLUSIONS:We concluded that MMP-9 gene polymorphisms may influence susceptibility to the development of chronic rhinosinusitis with nasal polyposis in Chinese population.
Intranasal or paranasal sinus olfactory neuroblastoma is a rare malignant neoplasm of olfactory neuroepithelial origin, accounting for approximately 5% of paranasal sinus cancers. Most of the presenting symptoms include nasal obstruction, nasal bleeding, anosmia, rhinorrhea, and headache. In this present report, we describe a 79-year-old man who presented with bilateral nasal congestion for more than 1 year. Nasoendoscopy showed a huge, smooth, mucosal tumor in the nasopharynx with extension to the posterior nasal septum. The tumor was completely resected under endonasal endoscopy and the pathology revealed olfactory neuroblastoma. Olfactory neuroblastomas usually arise in the cribriform plate and superior turbinate. However, the origin and isolation of olfactory neuroblastomas to the sphenoid sinus is exceedingly rare. Only four cases of olfactory neuroblastoma isolated in the sphenoid sinus have been described in English literature and the frequency of presenting symptoms with cranial neuropathies and headache. We report a case of primary sphenoid sinus olfactory neuroblastoma with the greatest enlargement reported to date.
OBJECTIVE Hypoxia may result in increased recruitment of inflammatory cells and release of various inflammatory cytokines. Local hypoxia within the sinus is believed to aggravate sinus inflammation. In this study, we tried to investigate the correlation of hypoxia‐inducible factor, which is upregulated under hypoxic condition, and nasal polyposis. SUBJECTS AND METHODS Forty‐four polyps from patients undergoing endoscopic sinus surgery and 38 inferior turbinate mucosa were obtained for immunohistochemical staining. Twenty‐three polyps and 22 turbinate mucosa also underwent real‐time reverse transcription‐polymerase chain reaction for quantification of messenger RNA (mRNA). The severity of the disease was judged by the Lund‐Mackay CT scoring. RESULTS Expression of hypoxia‐inducible factor protein, but not mRNA, was significantly increased in nasal polyps compared with that in the turbinate mucosa, but it did not correlate with the disease severity. CONCLUSION We suggest that hypoxia may have a role in the initiation of nasal polyposis.
Adenoid cystic carcinoma of the nasal septum is extremely rare. We present the case of a 56-year-old male who complained of nasal bleeding and nasal obstruction for 1 month. A mass arising from nasal septum was found by endoscope. The tumor was removed under lateral rhinotomy and histopathologic examination revealed adenoid cystic carcinoma with cribriform pattern. He then had postoperative radiotherapy. No recurrence was noticed after 1 year of follow-up. Despite its rarity, adenoid cystic carcinoma should be taken into consideration in the differential diagnosis of nasal tumor.
We report a young male patient who experienced seizure after local injection of 3 mL 2% lidocaine with epinephrine 1:200,000 around a recurrent nasal angiofibroma. After receiving 100% oxygen via mask and thiamylal sodium, the patient had no residual neurologic sequelae. Seizure immediately following the injection of local anesthetics in the nasal cavity is probably due to injection into venous or arterial circulation with retrograde flow to the brain circulation. Further imaging study or angiography should be done before head and neck surgeries, especially in such highly vascular neoplasm.