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1.  Idiopathic sclerosing inflammation presenting as sinusitis 
Allergy & Rhinology  2012;3(2):e101-e104.
Idiopathic sclerosing orbital inflammation is a rare finding that is poorly delineated, immune mediated, and causes severe symptoms and disability. It has been described affecting the orbit in addition to other sites within the head and neck, but has rarely been described presenting as sinusitis. A case report and literature review were performed. A 14-year-old girl with right-sided face and eye pain and pressure for >1 month presented 3 days after endoscopic sinus surgery for presumed acute sinusitis. She subsequently developed ipsilateral vision loss and hypesthesia of the infraorbital nerve. MRI revealed a mildly enhancing soft tissue intensity lesion extending from the right maxillary sinus into the pterygopalatine fossa and orbital apex through the inferior orbital fissure. Biopsy specimens of the lesion were consistent with a sclerosing inflammatory lesion. High-dose steroids led to rapid improvement in vision and pain; however, the patient was unable to tolerate steroid weaning because of recurrence of eye pain and headache. Repeat imaging showed progression of the lesion. Rheumatology was consulted and the patient's steroid therapy was altered and her medications were expanded to include azathioprine. The patient's symptoms improved and subsequent imaging showed a reduction in the size and extent of the lesion. Idiopathic sclerosing inflammation is characterized by primary, chronic, and immunologically mediated fibrosis. Patients typically have a poor response to corticosteroid treatment or radiotherapy. Immunosuppressive therapy in addition to corticosteroids is the recommended treatment.
PMCID: PMC3548607  PMID: 23342288
Azathioprine; extraorbital; idiopathic; inflammation; orbital; sclerosing; sinusitis
2.  Relationships between IL-17A and macrophages or MUC5AC in eosinophilic chronic rhinosinusitis and proposed pathological significance 
Allergy & Rhinology  2012;3(2):e50-e54.
Recently, some researchers have reported that macrophages and neutrophils were related to severe asthma. Mucus hypersecretion and persistent airway inflammation result from increased expression of mucin gene (MUC5AC). Eosinophilic chronic rhinosinusitis (ECRS) is considered as intractable rhinosinusitis. From the viewpoint of “one way one disease,” we examined whether ECRS is associated with infiltrating macrophages, neutrophils, their promotive factors, and MUC5AC. We examined 21 nasal polyps with CRS. Each specimen was fixed in 10% phosphate-buffered formalin, embedded in paraffin, processed routinely, and then prepared as semithin sections (3.5 μm). We immunohistochemically observed the macrophages by using CD68, neutrophils by using neutrophil elastase and the promotive factors, monocyte chemotactic protein (MCP) 1, IL-17A, and IL-8, in both ECRS and non-ECRS. The number of macrophages (CD68+ cells), IL-17A, and MUC5AC+ cells in ECRS were significantly greater than in non-ECRS. The mean number of MCP-1+ cells in ECRS was greater than that in non-ECRS, but not significantly. There was a significant correlation in all cases between IL-17A and macrophages or MUC5AC+ cells. Neither the numbers of neutrophils (positive cells for neutrophil elastase) nor the IL-8+ cells showed any significant differences between ECRS and non-ECRS. Our study suggested that infiltrating macrophages, IL-17A and MUC5AC, as well as eosinophils could have roles in the development of ECRS.
PMCID: PMC3548608  PMID: 23342289
Eosinophilic chronic rhinosinusitis; human; IL-8; IL-17A; macrophage; MCP-1; MUC5AC; nasal polyps; neutrophil
3.  The rate of epinephrine administration associated with allergy skin testing in a suburban allergy practice from 1997 to 2010 
Allergy & Rhinology  2012;3(2):e55-e60.
Allergy skin testing is considered a safe method for testing for IgE-mediated allergic responses although anaphylactic events can occur. Reported rates of anaphylaxis per patient are not consistent and range from 0.008 to 4%. The aim of this study was to determine the rate of epinephrine use associated with allergy skin-prick testing (SPT) and intradermal testing (IDT) in a suburban practice over 13 years. This retrospective chart review used billing and procedure coding records during the time period from January 1997 to June 2010 to identify encounters where epinephrine was administered after SPT or IDT. Patient encounters with procedure codes for skin testing plus either parenteral epinephrine, corticosteroid, antihistamine, or i.v. fluid administration were identified. These patient charts were reviewed to determine if epinephrine was administered, whether systemic reactions developed, and rates of epinephrine administration were calculated. There were 28,907 patient encounters for SPT and 18,212 for IDT. Epinephrine was administered in six patient encounters (0.02%) where SPT was performed; no IDT encounters led to epinephrine administration. There were no fatalities. Allergy skin testing to a variety of allergens, when administered by well-trained personnel, is a safe procedure. This study, involving the largest population to date, showed a rate of systemic reactions requiring epinephrine of 20 per 100,000 SPT visits. No epinephrine was given after IDT.
PMCID: PMC3548609  PMID: 23342290
Adverse reactions; allergic rhinitis; anaphylactoid reactions; anaphylaxis; food allergy testing; idiopathic anaphylaxis; intradermal testing; skin-prick testing; systemic reactions; venom allergy testing
4.  Measurement of osteotomy force during endoscopic sinus surgery 
Allergy & Rhinology  2012;3(2):e61-e65.
Greater understanding of the surgeon's task and skills are required to improve surgical technique and the effectiveness of training. Currently, neither the objective measurement of osteotomy forces during endoscopic sinus surgery (ESS) nor the validity of the properties of cadaver materials, are well documented. Measurement was performed of peak axial osteotomy force during ESS. A comparison was made of results with previously published cadaver data to validate the force properties of cadaver models. A prospective, consecutive cohort of 25 patients was compared with data from 15 cadaver heads. A modified Storz sinus curette measured osteotomy force from uncinate, bulla ethmoidalis, and ground lamella. Independent variables were osteotomy site, age, gender, indication for surgery, and side. Corresponding cadaver data were analyzed for the independent variables of osteotomy site, side, and gender and then compared with the live patient data. Mean osteotomy force in live patients was 9.6 N (95% CI, 8.9–10.4 N). Mean osteotomy force in the cadaver heads was 6.4 N (95% CI, 5.7–7.0 N). Ethmoid osteotomy of live patients required 3.2 N (95% CI, 2.1–4.3 N) more force than the cadaver heads (p = 0.0001). This relationship was statistically significant at the bulla ethmoidalis (p = 0.002) and the ground lamella (p = 0.0001) but not at the uncinate (p = 0.068). Osteotomy in female live subjects required 1.6 N (95% CI, 0.1–3.1 N) more force than male live subjects (p = 0.03). Cadaver tissue may underestimate the mean osteotomy force required in osteotomy of living ethmoid sinus lamellae by a factor of 1.5 times. Caution may be required in extrapolating force estimates from cadaver tissue to those required in living patients.
PMCID: PMC3548610  PMID: 23342291
Biomechanics; curette; endoscopic surgical procedures; ergonomics; force; measurement techniques; osteotomy; otolaryngology; paranasal sinuses; surgical therapy
5.  Diffusion lung capacity of carbon monoxide: A novel marker of airways remodeling in asthmatic children? 
Allergy & Rhinology  2012;3(2):e66-e73.
Asthma is universally considered a chronic inflammatory disorder of the airways. Several noninvasive markers, such as exhaled nitric oxide (FeNO) and exhaled breath temperature (PletM), have been proposed to evaluate the degree of airway inflammation and remodeling in asthmatic children. The aim of this study was to evaluate the relationship between diffusion lung capacity of carbon monoxide (DLCO) and these inflammatory markers in asthmatic children. We compared data of FeNO, PletM, and DLCO collected in 35 asthmatic children at admission (T0) and discharge (T1) after a period spent in a dust-mite–free environment (Misurina, Italian Dolomites, 1756 m). PletM showed a reduction from 29.48°C at T0 to 29.13°C at T1 (p = 0.17); DLCO passed from 93 to 102 (p = 0.085). FeNO mean value was 29.7 ppb at admission and 18.9 ppb at discharge (p = 0.014). Eosinophil mean count in induced sputum was 4 at T0 and 2 at T1 (p = 0.004). Spearman standardization coefficient beta was 0.414 between eosinophils and FeNO and −0.278 between eosinophils and DLCO. Pearson's correlation index between DLCO and PletM was −0.456 (p = 0.019). A negative correlation between DLCO and PletM was found. However, DLCO did not show a significant correlation with FeNO and eosinophils in the airways. Additional studies are needed to clarify the role of DLCO as a potential tool in monitoring childhood asthma.
PMCID: PMC3548611  PMID: 23342292
Breath temperature; childhood asthma; diffusion lung capacity; exhaled nitric oxide; inflammation; remodeling; sputum eosinophils
6.  Group 10 allergens (tropomyosins) from house-dust mites may cause covariation of sensitization to allergens from other invertebrates 
Allergy & Rhinology  2012;3(2):e74-e90.
Group 10 allergens (tropomyosins) have been assumed to be a major cause of cross-reactivity between house-dust mites (HDMs) and other invertebrates. Despite all of the published data regarding the epidemiology, percent IgE binding and level of sensitization in the population, the role of tropomyosin as a cross-reactive allergen in patients with multiple allergy syndrome still remains to be elucidated. Homology between amino acid sequences reported in allergen databases of selected invertebrate tropomyosins was determined with Der f 10 as the reference allergen. The 66.9 and 54.4% identities were found with selected crustacean and insect species, respectively, whereas only 20.4% identity was seen with mollusks. A similar analysis was performed using reported B-cell IgE-binding epitopes from Met e1 (shrimp allergen) and Bla g7 (cockroach allergen) with other invertebrate tropomyosins. The percent identity in linear sequences was higher than 35% in mites, crustaceans, and cockroaches. The polar and hydrophobic regions in these groups were highly conserved. These findings suggest that tropomyosin may be a major cause of covariation of sensitization between HDMs, crustaceans, and some species of insects and mollusks.
PMCID: PMC3548612  PMID: 23342293
Cross-reactivity of tropomyosins; group 10 allergens; HDM allergens; homology; IgE-binding epitopes; multiple allergy syndrome; tropomyosins
7.  Cadaveric analysis of nasal valve suspension 
Allergy & Rhinology  2012;3(2):e91-e93.
This study was designed to measure the efficacy of a nasal valve suspension technique and determine the adequate traction length without creation of nasofacial fullness in a cadaveric model. Seven fresh frozen cadaveric heads were evaluated. Minimal cross-sectional (MCA) areas were measured with a transient-signal acoustic rhinometer (Ecco Vision; Hood Instruments, Pembroke, MA) before and after suspension. The adequate traction length, which did not cause obvious changes, was determined. Five millimeters of lateral nasal valve traction was determined to be the maximal traction achievable without creating facial fullness. After lateral nasal suspension, average MCA increased by 13.7%. Average distance to the MCA from the nostril changed from 1.57 to 1.76 cm. Postsuspension values were significantly higher than the presuspension values (p < 0.05). Nasal valve suspension with 5 mm of lateral traction has a significant impact on nasal valve area without obvious nasofacial changes.
PMCID: PMC3548613  PMID: 23342294
Acoustic rhinometry; analysis; cadaver; complication; nasal obstruction; nasal valve; stenosis; surgery; suspension; traction length
8.  Signal transducer and activator of transcription 3 mutation with invasive eosinophilic disease 
Allergy & Rhinology  2012;3(2):e94-e97.
Hyper-IgE syndrome (HIES), or Jobs disease, is a rare immunologic disorder characterized by the triad of staphylococcal abscesses, pneumonia with pneumatocele formation, and elevated IgE. It has been shown to have multiple modes of inheritance, autosomal dominant being more common than autosomal recessive, with sporadic cases as well. A mutation in signal transducer and activator of transcription 3 (STAT3) gene has been linked to the development of the sporadic and dominant forms of HIES. Peripheral eosinophilia, typically greater than two standard deviations from the normal population, is often seen in association with HIES. Despite these elevated levels of blood eosinophils, there have been no reported cases of invasive eosinophilic disease, such as eosonophilic esophagitic. Here we report the first description, to our knowledge, of a patient with HIES with a STAT3 mutation involving exon 12, Thr389Ile, and invasive eosinophilic disease of the esophagus. STAT3 modulates the expression of several genes that control central cell processes such as growth and death in response to external soluble stimuli. A mutation in the STAT3 molecule may affect the eosinophil's response to IL-5 and thus reduce the chemotaxic ability of those cells to migrate into tissues. This may then explain the paucity of eosinophilic infiltrative disease in patients with STAT3 mutations. The level of eosinophilic involvement may be related to the site or type of mutation within the STAT3 molecule. As more data are collected, we may be able to assess whether certain mutations dictate different clinical outcomes, which could prove helpful in directing therapy.
PMCID: PMC3548614  PMID: 23342295
Eosinophilia; esophagitis; hyper-IgE; immunodeficiency; invasive; STAT3; Thr389Ile
9.  Displaced nasal dilator caused severe pain: Case report and literature review 
Allergy & Rhinology  2012;3(2):e98-e100.
Internal nasal dilators are widely used but have not been reported to cause severe symptoms. We describe a case in which a male adult had accidentally, during sleep, inhaled a nasal dilator into his right nasal cavity, and we review the relevant literature. A PubMed search was performed of nasal dilators, especially of the internal types, including “Nasaline Snooze'” (ENTPro, Stockholm, Sweden). A foreign body in adults may be an inhaled nasal dilator. It may be overlooked on computed tomography scans, and thorough inspection of the nose is diagnostic.
PMCID: PMC3548615  PMID: 23342296
Adult; computed tomography; foreign body; head ache; nasal dilator; nasal obstruction; sinusitis
10.  Quality of life comparison in common rhinologic surgeries 
Allergy & Rhinology  2012;3(1):e1-e7.
Various questionnaires are used in patients who undergo rhinologic surgeries but a unique comprehensive questionnaire is needed to evaluate quality of life (QOL) in rhinologic surgeries. The purpose of this study was to prepare a comprehensive questionnaire and compare QOL among four common rhinologic surgeries including functional endoscopic sinus surgery, septoplasty, septorhinoplasty, and septoplasty with turbinoplasty preoperatively and 6 months postoperatively. This was a prospective interventional before-and-after study. Preoperative and 6 months postoperative evaluations were performed with a Modified Health-Related Quality of Life (HRQL) questionnaire designed to cover all needed QOL aspects and the 22-item Sino-nasal Outcome Test questionnaire to cover all needed QOL aspects. The Modified HRQL included 33 items in six subgroups (nasal symptoms, sleep problems, headache, nonnasal symptoms, and practical and emotional problems) and general feeling. From 202 patients who completed the questionnaire before the procedures, 146 (72% of all patients) who were interviewed 6 months postoperatively were included in this study. Comparing preoperative data between followed up patients and missed patients showed no statistical difference among surgeries (p = 0.90). Comparison of patient's pre- and postoperative QOL showed a significant improvement in global QOL and in all questionnaire items (p < 0.0001 in all comparisons). Comparison of QOL changes before and after surgery among different surgeries revealed no statistical difference (p = 0.282). Our data showed a significant improvement in each surgery but the amount of improvement in different surgeries was almost constant.
PMCID: PMC3404471  PMID: 22852123
Functional endoscopic sinus surgery; Modified Health-Related Quality of Life questionnaire; quality of life; rhinologic surgeries; septoplasty; septorhinoplasty; turbinoplasty
11.  Rhinologic issues in pregnancy 
Allergy & Rhinology  2012;3(1):e13-e15.
The diagnosis and treatment of rhinitis, sinusitis, and epistaxis during pregnancy present unique challenges to the otolaryngologist. Poorly controlled sinonasal disease may have significant adverse effects on the mother's quality of life and pregnancy outcomes and the lack of adequately controlled safety data limits the clinician's ability to make informed decisions about management. At the conclusion of this discussion, the reader should be familiar with the available literature and evidence-based guidelines regarding the safety and indications for radiographic imaging, clinical testing, medical intervention, and surgical treatment of sinonasal disease in pregnant patients. A review was performed of pertinent guidelines regarding the management of gestational rhinitis, sinusitis, and epistaxis, including the diagnostic and therapeutic limitations and physiological changes specific to pregnancy. A study population of four patients was analyzed to highlight the steps of management by reviewing the patient charts including pertinent history, physical examination, clinical course, and operative reports. Two patients with epistaxis and two patients with rhinosinusitis ranging from 27 to 38 years of age and between 16 and 35 weeks gestation were analyzed. The treatment of sinonasal disease during pregnancy is challenging and a thorough knowledge of the available medical evidence and treatment guidelines is necessary to optimize pregnancy outcomes. When the severity of disease precludes the possibility of delaying treatment, the clinician should provide a limited intervention that optimizes the mother's health without placing the fetus at significant risk.
PMCID: PMC3404472  PMID: 22852124
Advair; albuterol; allergic rhinitis; amoxicillin; anaphylaxis; Augmentin; azithromycin; budesonide; epistaxis; fluticasone; gestational rhinitis; montelukast; prednisone; pyogenic granuloma; rads; rhinitis medicamentosa; rhinitis of pregnancy; sinusitis
12.  Sensitization rate and clinical profile of Congolese patients with rhinitis 
Allergy & Rhinology  2012;3(1):e16-e24.
In the African continent, the sensitization pattern and clinical profile are unknown in patients with rhinitis/rhinosinusitis attending the outpatient ear, nose, and throat (ENT) clinics. We therefore aimed to analyze the clinical characteristics of rhinitis/rhinosinusitis patients in Democratic Republic of Congo (DRC), classify allergic rhinitis (AR) according to the Allergic Rhinitis and Its Impact on Asthma criteria, and evaluate the sensitization profile and its associated factors. From January to May 2009, 423 patients with rhinitis symptoms attending the Outpatient ENT clinic of the University Hospital and Saint Joseph Hospital of Kinshasa were evaluated for allergy symptoms, severity, and duration of symptoms and underwent skin-prick tests (SPTs) for a panel of 15 allergens. Of 423 patients 35.2% had positive SPT results, with 40.9% showing polysensitization. Dermatophagoides pteronyssinus (DPT) (68.5%) and cockroach (36.2%) were the most common allergens among sensitized patients. Patients with rhinitis/rhinosinusitis mainly presented in decreasing order with sneezing, facial pain/pressure, nasal obstruction, postnasal discharge, nose itching, clear nasal discharge, and eye itching. Persistent and moderate/severe AR represented 61.4 and 69.3%, respectively. Sensitization was independently associated with younger age, rhinoconjunctivitis, and reaction to nonspecific trigger factors. In conclusion, 35.2% of patients attending the ENT Outpatient Clinic in DRC for rhinitis problems had a positive SPT to at least one allergen, with mainly DPT and cockroach allergens being involved; and a substantial portion showed persistent and moderate/severe AR. Therefore, allergy should not be neglected as an etiologic factor in rhinologic disease in the African continent.
PMCID: PMC3404473  PMID: 22852125
Congo; rhinitis; rhinosinusitis; skin-prick testing; symptoms
13.  Mite-induced inflammation: More than allergy 
Allergy & Rhinology  2012;3(1):e25-e29.
Clinical observations have suggested that there is an association of atopic conditions with hypersensitivity reactions to nonsteroidal anti-inflammatory drugs (NSAIDs). This relationship has been especially present in patients allergic to mites. This study was designed to review clinical and experimental evidence linking atopy, mite allergy, and hypersensitivity to aspirin and NSAIDs and discuss the possible mechanisms explaining this association. A review of the medical literature concerning the association of atopic diseases, mite hypersensitivity, and intolerance to NSAIDs using PubMed and other relevant articles is presented. NSAID-sensitive patients are frequently atopic and allergic to mites, and patients who develop oral mite anaphylaxis (OMA) show an increased prevalence of NSAID hypersensitivity. The study of atopic, mite-sensitive patients, who experience urticaria and angioedema when exposed to NSAIDs and patients with OMA suggests an interesting interaction between atopic allergy and disorders of leukotriene synthesis or metabolism. Various mechanisms that could be involved in this interaction are presented, including genetic factors, inhibition of cyclooxygenase-1, and other effects (not related to IgE sensitization) of mite constituents on the immune system. The association of mite hypersensitivity with aspirin/NSAIDs intolerance has been confirmed and provides additional clues to various nonallergic pathways that may contribute to the acute and chronic inflammatory process observed in atopic, mite-allergic, individuals. The clinical relevance of these observations is presently under investigation.
PMCID: PMC3404474  PMID: 22852126
Aspirin; acetylsalicylic acid; angioedema; cysteinyl-leukotrienes; Dermatophagoides; immunoglobulin E; mites; leukotriene C4 synthase; nonsteroidal anti-inflammatory drugs; NSAIDs
14.  Regulation of dendritic cell functions against harmful respiratory pathogens by a cysteinyl leukotrienes receptor antagonist 
Allergy & Rhinology  2012;3(1):e30-e34.
Cysteinyl leukotriene receptor antagonist (LTRA) is a widely used medicine for asthma. Cysteinyl leukotrienes (cysLTs) are involved in the regulation of dendritic cell (DC) function. However, the effects of LTRA on DC-related antimicrobial immunity against harmful respiratory pathogens remain unknown. The purpose of this study was to examine the effects of LTRA administered in vivo on DC function against representative respiratory pathogens in vitro. Pulmonary DCs were isolated from four groups of mice: control, mite allergen sensitized (AS), and AS mice treated with the corticosteroid dexamethasone (Dex) or with the LTRA pranlukast (Prl). These DCs were incubated with mite allergen, lipopolysaccharide (LPS), Aspergillus fumigatus, or respiratory syncytial virus (RSV). IL-10 and IL-12 production was then determined. Dex treatment significantly inhibited lipopolysaccharide (LPS)-induced IL-10 and IL-12 production as well as baseline IL-12 production in AS mice. The Prl did not significantly inhibit LPS-induced IL-10 and IL-12 production in AS mice. More importantly, Prl significantly increased IL-10 and IL-12 in AS mice after RSV infection. This study shows that LTRA that is used for asthma potentially up-regulates antimicrobial immunity through modulation of DC function against some respiratory infections without immunosuppression.
PMCID: PMC3404475  PMID: 22852127
Allergic airway inflammation; Aspergillus fumigatus; asthma; corticosteroids; cysteinyl leukotrienes receptor antagonist; cytokines; dendritic cell; Dermatophagoides farinae; lipopolysaccharide; respiratory syncytial virus
15.  Polymorphic variants of interleukin-13 R130Q, interleukin-4 T589C, interleukin-4RA I50V, and interleukin-4RA Q576R in allergic rhinitis: A pilot study 
Allergy & Rhinology  2012;3(1):e35-e40.
The development of allergic rhinitis is considered to be caused by the complex interactions between genetic predisposition and environmental factors. Polymorphisms in the interleukin (IL)-13/4/4RA pathway have previously been shown to be associated with atopic diseases. The purpose of this study was to determine the association between IL-13 R130Q, IL-4 T589C, IL4 receptor alpha (IL-4RA) I50V, or IL-4RA Q576R polymorphisms and risk of allergic rhinitis in a hospital-based Malaysian population. A case-control pilot study was undertaken and genotyping of these polymorphisms was performed using polymerase chain reaction–restriction fragment length polymorphism on 54 allergic rhinitis patients and 45 healthy individuals. Polymorphism of IL-13 R130Q showed significant difference in genotype (p = 0.048) and allele (p = 0.002) frequencies in allergic rhinitis when compared with healthy controls. Individuals who were GA heterozygotes (adjusted odds ratio [ORadj] = 3.567; 95% CI, 1.211–10.509), and carriers of A allele genotype (ORadj = 3.686; 95% CI, 1.300–10.451) and A allele (ORadj = 3.071; 95% CI, 1.514–6.232) had an elevated risk of developing allergic rhinitis. The genotype and allele frequencies of IL-4 T589C, IL-4RA I50V, and IL-4RA Q576R polymorphisms were not significantly different between the allergic rhinitis patients and normal healthy individuals and did not show an associated risk with allergic rhinitis. Our findings indicate that polymorphic variant of IL-13 R130Q appears to be associated with increased risk for development of allergic rhinitis in a hospital-based Malaysian population but not IL-4 T589C, IL-4RA I50V, and IL-4RA Q576 polymorphisms. Additional studies using larger sample size are required to confirm our findings and its exact role in allergic rhinitis.
PMCID: PMC3404476  PMID: 22852128
Allele; allergy; RFLP; genotyping; IL-13; IL-4; IL-4RA; PCR; polymorphism; rhinitis
16.  Locally destructive skull base lesion: IgG4-related sclerosing disease 
Allergy & Rhinology  2012;3(1):e41-e45.
A unique case of IgG4+ sclerosing disease was diagnosed in the sphenoid sinus, a previously unreported location, and was treated in a novel manner. This study describes the clinical presentation and management of IgG4 sclerosing disease in the paranasal sinuses. A retrospective case review and review of the medical literature were performed. A 38-year-old woman with a 2-year history of constant frontal headaches presented to our clinic. Imaging showed bony destruction of the sphenoid sinus and sellar floor. The patient underwent a right-sided sphenoidotomy with debridement and biopsy. Pathological evaluation showed a dense plasmacytic infiltrate with >150 IgG4+ cells/high-power field. She was subsequently started on a nasal corticosteroid with improved patency of the sphenoid antrostomy. We report an unusual case of a middle-aged woman who presented with IgG4-sclerosing disease (IGSD) isolated to the sphenoid sinus. Although our knowledge concerning treatment in extrapancreatic organs is lacking, there is evidence that glucocorticoid treatment improves nasal sinus opacification on CT findings (Sato Y, Ohshima K, Ichimura K, et al., Ocular adnexal IgG4-related disease has uniform clinicopathology, Pathol Int 58:465–470, 2008). This case study and literature review adds to the growing literature describing IGSD in the head and neck and more specifically isolated to the sphenoid sinus with preliminary data concerning local control with topical steroids.
PMCID: PMC3404477  PMID: 22852129
Corticosteroid; IgG4; IGSD; sclerosing disease; skull base; sphenoid; sphenoidectomy
17.  Congenital nasolacrimal duct cyst/dacryocystocele: An argument for a genetic basis 
Allergy & Rhinology  2012;3(1):e46-e49.
Embryogenesis of a congenital nasolacrimal duct (NLD) cyst is attributed to the failure of the Hasner membrane of the NLD system to cannulate. Prenatal diagnosis of congenital NLD cysts supports the argument for a developmental error, with a postnatal prevalence of 6%. The role of a genetic basis for this malformation has never been ascribed. We present a set of monozygotic twins with bilateral congenital NLD cysts as an argument for a genetic basis of this entity. A case report and literature review were performed. We present two cases of bilateral congenital NLD cysts occurring in a set of monozygotic twins. Patients were delivered at 37 weeks via cesarean section. The pregnancy was complicated by preterm labor at 33 weeks requiring administration of terbutaline and betamethasone. At presentation, twin A had bilateral eye discharge, erythema, and swelling medial to the medial canthi as well as nasal obstruction. Computed tomography (CT) showed classic bilateral cystic masses in the inferior meatus. The diagnosis of bilateral infected congenital dacryocystoceles was made. Twin B initially presented with only bilateral eye discharge and CT showed a dilated NLD system. Twin B subsequently developed early signs of bilateral dacryocystoceles the following day. Both patients underwent lacrimal probing and endoscopic marsupialization of the dacryocystoceles. Biopsies were consistent with dacryocystocele. Dacryocystocele is a common presentation of unresolved neonatal NLD obstruction. This case report in a set of identical twins is an argument for a genetic basis for the formation of this lesion.
PMCID: PMC3404478  PMID: 22852130
Congenital; cyst; dacryocystocele; dacryocystorhinostomy; genetic; Hasner; nasolacrimal
18.  Efficacy of medical therapy in treatment of chronic rhinosinusitis 
Allergy & Rhinology  2012;3(1):e8-e12.
Uncomplicated chronic rhinosinusitis (CRS) is generally treated with medical therapy initially and surgery is contemplated only after medical therapy has failed. However, there is considerable variation in the medical treatment regimens used and studies defining their efficacy are few. The aim of this study was to determine the proportion of patients treated medically who responded sufficiently well so that surgery was not required. Subgroup analysis to identify clinical features that predicted a favorable response to medical therapy was also performed. Eighty patients referred to the Otorhinolaryngology Clinic at North Shore Hospital were treated with a standardized medical therapy protocol (oral prednisone for 3 weeks, oral antibiotics and ongoing saline lavage and intranasal budesonide spray). Symptom scores were collected before and after medical therapy. Clinical features such as presence of polyps, asthma, and aspirin hypersensitivity were recorded. Failure of medical therapy was defined as the persistence of significant CRS symptoms, and those patients who failed medical therapy were offered surgery. Follow-up data were available for 72 (90%) patients. Of this group, 52.5%, (95% CI, 42.7%, 62.2%) failed to respond adequately to medical therapy and were offered surgery. The remaining patients (37.5%) were successfully treated with medical therapy and did not require surgery at the time of follow-up. The premedical therapy symptom scores were significantly higher than the postmedical therapy symptom scores (p < 0.01). The symptom scores of those patients postmedical therapy who proceeded to have surgery were significantly higher than the group who responded well to maximum medical therapy (MMT) and did not require surgery (p < 0.0001). There were no significant differences in the proportion of patients with asthma, aspirin sensitivity, or polyps between the groups failing or not failing MMT. In approximately one-third of patients with CRS, medical therapy improved symptoms sufficiently so that surgical therapy was avoided. Patients with more severe symptoms tended not to respond as well as those with less severe symptoms. Long-term follow-up is required for the group of responders to determine how many will eventually relapse.
PMCID: PMC3404479  PMID: 22852131
Antibiotics; chronic rhinosinusitis; corticosteroid; intranasal steroid; macrolides; maximal medical therapy; medical therapy; prednisone; saline irrigation; symptom

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