Mucocele Formation Research Articles

A CASE REPORT OF RECURRENT FRONTAL MUCOCOELE

Background: Paranasal sinus mucocele is the epithelium lined mucus filled sac causing enlargement of sinus wall. It can cause bone erosion and displacement of surrounding structures. Mucoceles usually occur due to infection, inflammation, trauma, previous surgeries, and tumours which can potentially obstruct the sinus ostium. Cranio facial trauma is the commonest cause for mucocele formation. Case report: A 50yr old male patient came with chief complaints of swelling on forehead since 1yr. The patient had history of road traffic accident 13yrs ago for which he underwent facio-maxillary reconstruction along with right eye evisceration. Later he developed a swelling above right eye after 7yrs, which was diagnosed as mucocele and was treated by frontal cranioplasty. Now he came with complaint of swelling on forehead which was gradually increasing in size. MRI-PNS was done showing fluid filled space extending anteriorly from cutaneous plane in the frontal region going posteriorly over the orbit pushing the contents of the orbit inferiorly and medially and extending posteriorly up to orbital apex. Patient underwent endoscopic mucocele drainage and DRAF 3. Right orbital exenteration was done to prevent recurrence. Patient was on follow up since then and no symptoms or signs of recurrence is noted. Conclusion: Facial trauma may be one of the reasons for development of mucocele where sinus drainage pathways are deranged. As in our case trauma not only led to development of mucocele, but new spaces also created by the treatment lead to extensive spread of the mucocele.

Acquired dysfunction of CFTR underlies cystic fibrosis-like disease of the canine gallbladder.

Mucocele formation in dogs is a unique and enigmatic muco-obstructive disease of the gallbladder caused by the amassment of abnormal mucus that bears striking pathological similarity to cystic fibrosis. We investigated the role of cystic fibrosis transmembrane conductance regulatory protein (CFTR) in the pathogenesis of this disease. The location and frequency of disease-associated variants in the coding region of CFTR were compared using whole genome sequence data from 2,642 dogs representing breeds at low-risk, high-risk, or with confirmed disease. Expression, localization, and ion transport activity of CFTR were quantified in control and mucocele gallbladders by NanoString, Western blotting, immunofluorescence imaging, and studies in Ussing chambers. Our results establish a significant loss of CFTR-dependent anion secretion by mucocele gallbladder mucosa. A significantly lower quantity of CFTR protein was demonstrated relative to E-cadherin in mucocele compared with control gallbladder mucosa. Immunofluorescence identified CFTR along the apical membrane of epithelial cells in control gallbladders but not in mucocele gallbladder epithelium. Decreases in mRNA copy number for CFTR were accompanied by decreases in mRNA for the Cl-/[Formula: see text] exchanger SLC26A3, K+ channels (KCNQ1, KCNN4), and vasoactive intestinal polypeptide receptor (VIPR1), which suggest a driving force for change in secretory function of gallbladder epithelial cells in the pathogenesis of mucocele formation. There were no significant differences in CFTR gene variant frequency, type, or predicted impact comparing low-risk, high-risk, and definitively diagnosed groups of dogs. This study describes a unique, naturally occurring muco-obstructive disease of the canine gallbladder, with uncanny similarity to cystic fibrosis, and driven by the underlying failure of CFTR function.NEW & NOTEWORTHY Cystic fibrosis transmembrane conductance regulatory protein (CFTR) genomic variants and expression of mRNA, protein, and electrogenic anion secretory activity of CFTR were characterized in dog gallbladder. Acquired inhibition of CFTR expression by gallbladder epithelium was identified as underpinning a naturally occurring muco-obstructive disease of the dog gallbladder that bears striking pathological similarity to animal models of cystic fibrosis.

Increased lipogenesis and lipidosis of gallbladder epithelium in dogs with gallbladder mucocele formation.

Gallbladder disease in people is frequently associated with disorders of lipid metabolism and metabolic syndrome. A recently emergent gallbladder disease of dogs, referred to as mucocele formation, is characterized by secretion of abnormal mucus by the gallbladder epithelium and is similarly associated with hyperlipidemia, endocrinopathy, and metabolic dysfunction. The cause of gallbladder mucocele formation in dogs is unknown. A prospective case-controlled study was conducted to gain insight into disease pathogenesis by characterization of plasma lipid abnormalities in 18 dogs with gallbladder mucocele formation and 18 age and breed matched control dogs using direct infusion mass spectrometry for complex plasma lipid analysis. This analysis was complemented by histochemical and ultrastructural examination of gallbladder mucosa from dogs with gallbladder mucocele formation and control dogs for evidence of altered lipid homeostasis of the gallbladder epithelium. Gallbladder mucocele formation in dogs carried a unique lipidomic signature of increased lipogenesis impacting 50% of lipid classes, 36% of esterified fatty acid species, and 11% of complex lipid species. Broad enrichment of complex lipids with palmitoleic acid (16:1) and decreased abundance within complex lipids of presumptive omega-3 fatty acids eicosapentaenoic (20:5) and docosahexaenoic (22:6) was significant. Severe lipidosis of gallbladder epithelium pinpoints the gallbladder as involved causally or consequently in abnormal lipid metabolism. Our study supports a primary increase in lipogenesis in dogs with mucocele formation and abnormal gallbladder lipid metabolism in disease pathogenesis.

Use of Titanium Mesh During Frontal Sinus Setback in Facial Feminization Surgery: Clinical Outcomes and Perspectives.

Brow position, hairline shape, and forehead projection may confer cis-feminine identity, and facial feminization surgery (FFS) can improve gender dysphoria among transfeminine patients. Depending on the Ousterhout classification, burring, osteotomy, anterior frontal sinus setback, and augmentation, with subsequent fixation via metal or bioabsorbable plates, can address forehead projection. However, titanium mesh, often used in frontal sinus fracture repair, has not been described for forehead contouring in FFS. The purpose of this study was to study clinical outcomes associated with the use of titanium mesh for the stabilization of bone following anterior frontal sinus setback. A retrospective cohort study of trans-female and nonbinary patients undergoing primary FFS by our senior author between January 2021 and February 2023 was performed. Variables collected include demographics, Ousterhout classification, operative details, complications, and follow-up duration. Patients with prior FFS or facial trauma were excluded. Data were analyzed using SPSS, (IBM, Armonk, NY). Forty-three transfeminine patients were included for analysis. The cohort had an average age of 33.0±8.7 years and a median follow-up time of 3.0 months (IQR = 1.0-7.0). Among our cohort, 26 patients (60.5%) received titanium mesh and 17 patients (39.5%) underwent burring only for forehead contouring. There were no reported complications (ie, infection, hardware extrusion, or mucocele formation) or a need for forehead revisions among the entire cohort regardless of the frontal sinus reconstruction material utilized. Clinical outcomes were favorable among patients receiving titanium mesh anterior table fixation during FFS. Titanium mesh can be considered as an additional technique for frontal bossing reduction and anterior table fixation in FFS.

Frontal sinus reconstruction with overlapping sinus mucosa and vascularized pericranial flap Coverage after modified transbasal bifrontal craniotomy: Novel technique and clinical outcomes

BackgroundThe modified transbasal bifrontal craniotomy is a variant of the bifrontal craniotomy with a wider surgical corridor than the standard approach. There are several methods for frontal sinus repair in bifrontal craniotomy. This study reports a novel method for frontal sinus repair in the modified transbasal interhemispheric approach by precisely overlapping the frontal sinus mucosa margin (without frontal sinus mucosa exenteration) with packing the frontal sinus with povidone-soaked gel foam and covering it with a vascularized pericranial flap. MethodsIn this case series, we retrospectively collected the clinical outcomes regarding cerebrospinal fluid (CSF) leakage, meningitis, and mucocele formation of patients who underwent modified transbasal bifrontal craniotomy at Vara Hospital. ResultsFrom January 2016 to December 2021, 65 patients with anterior skull-base lesions were treated with a modified transbasal interhemispheric approach with frontal sinus repair by overlapping frontal sinus mucosa with gel foam packing and vascularized pericranium flap covering. There was no case of postoperative CSF leakage, meningitis, or mucocele formation during the follow-up period of 19.2 months (min 1, max 73). ConclusionsWe demonstrated that the modified transbasal interhemispheric approach with frontal sinus repair using gel foam packing and pericranial flap is effective in preventing postoperative CSF leakage and meningitis.

Functional and Esthetic Outcomes of Either Surgically or Conservatively Treated Anterior Frontal Sinus Wall Fractures: A Long-Term Follow-Up

Study Design Retrospective cohort study. Objective Frontal sinus fractures (FSFs) can lead to a range of clinical challenges, including facial deformity, impaired facial sensation, cerebrospinal fluid (CSF) leakage, sinus drainage impairment, chronic sinus pain and mucocele formation. The optimal management approach, whether surgical or conservative, remains a topic of ongoing discussion. The aim of this study was to evaluate and compare the functional and esthetic outcomes of patients with surgically and conservatively treated FSFs. Methods In this retrospective study, patients treated for FSFs at the Karolinska university hospital 2004 to 2020 were identified in hospital records and invited to participate in a long-term follow-up. Sequelae and satisfaction with the esthetic result were assessed trough questionnaires and physical examinations. Results A total of 93 patients were included in the study, with 49 presenting isolated anterior wall fractures and 44 presenting combined anterior and posterior wall fractures. Surgical intervention was performed in 45 cases, while 48 were managed conservatively. Among patients with moderate anterior wall fractures (4-6 mm dislocation), 80% of surgically treated patients compared to 100% of conservatively treated patients expressed satisfaction with their cosmetic outcomes at follow-up ( P = .03). In conservatively treated patients with a forehead impression, the anterior wall fracture dislocation ranged from 5.3 to 6.0 mm ( P < .0001). Approximately 50% of surgically treated patients vs 15% of conservatively treated patients developed impaired forehead sensation at follow-up ( P = .03). Thirty-six percent of surgically treated patients reported dissatisfaction with surgery-related scarring, particularly those who underwent surgery via laceration or bicoronal incision. Conclusions This study suggests that anterior FSFs with a dislocation of 5 mm or less can be effectively managed conservatively with high patient satisfaction, low risk of long-term forehead sensation impairment and without potential development of forehead impression. Bicoronal incision or incision via a laceration may be associated with esthetic dissatisfaction and late sequelae such as alopecia.

Orbital Reconstruction Using a Polyetheretherketone Patient-Specific Implant After Removal of a Mucocele Developing After Orbital Fracture Repair.

A few mucoceles developing secondary to facial bone fractures have been reported. Mucocele formation is thought to be attributable to displacement of the respiratory mucosa with obstruction of the sinus opening, especially if untreated. Accurate diagnosis and management are required; a growing mucocele will gradually destroy adjacent bony structures and cause irreversible complications. The authors describe a patient who presented with diplopia and mild discomfort when gazing upward. She had undergone reconstruction of medial and inferior orbital fractures 20 years previously. The patient was diagnosed with a mucocele developing after orbital fracture repair. The patient underwent mucocele removal and orbital reconstruction using a polyetheretherketone patient-specific implant. In a patient with orbital symptoms but without acute trauma, a mucocele should be among the differential diagnoses if history-taking reveals past orbital trauma and surgery. A polyetheretherketone patient-specific implant was effective for orbital reconstruction after mucocele removal.

Autologous Fat Versus Hydroxyapatite Cement for Use in Frontal Sinus Obliteration: Systematic Review and Meta-Analysis.

Frontal sinus obliteration is a possible terminus for the management of chronic sinusitis, frontal bone trauma, cancer extirpation, or mucocele. The mucosa of the sinus is stripped and space obliterated with either autogenic, allopathic, or synthetic materials. This study aimed to compare the outcomes of autologous fat and hydroxyapatite cement (HAC) for frontal sinus obliteration. A multidatabase systematic review was conducted to collect outcomes on frontal sinus obliterations with either autologous fat or HAC. Outcomes collected included infection of the frontal sinus or obliteration material, mucocele formation, skin infection at the operative site, hematoma or seroma, cosmetic defect, donor site infection, and need for revision procedure. Prevalence of outcomes was analyzed through meta-analysis of proportions. Twenty studies were ultimately included in the meta-analysis. Sixteen studies reported outcomes for 667 patients obliterated with autologous fat, and 7 studies reported outcomes for 100 patients obliterated with HAC. Prevalence of infection of the frontal sinus or obliteration material, mucocele formation, skin infection at the operative site, hematoma or seroma, and cosmetic defect did not differ statistically between the cohorts. Donor site infection secondary to fat harvesting for fat obliteration had a prevalence of <0.01. Revision rate was 0.04 (95% confidence interval, 0.01-0.8) for fat obliteration and 0.10 (95% confidence interval, 0.00-0.33) for HAC obliteration. No statistically significant differences in the studied outcomes were detected between obliterations with autologous fat and HAC, suggesting that either is equally suitable and may be up to the surgeon's preference.

Inflammatory Sinonasal Polyposis-Causing Globe Perforation-With Response to Dupilumab Therapy.

A 75-year-old male patient with asthma presented with left eye vision loss for 1 year and progressive nasal obstruction, anosmia, and purulent eyelid and nasal discharge for several months. There was no light perception vision with an afferent pupillary defect in the left eye. Examination demonstrated nasal dorsum widening, sino-cutaneous fistulae, bilateral proptosis, and left eye chemosis with corneal keratinization in the setting of prior perforation (Fig. 1A). Nasal endoscopy showed severe, bilateral polyposis (Fig. 1B). MRI revealed an extensive sinonasal process with left globe disfigurement and severe, postobstructive, sinus mucocele formation (Fig. 2, top). The patient underwent bilateral functional endoscopic sinus surgery. Intraoperatively extensive sinonasal polyposis and inspissated mucinous debris were encountered. Histopathology demonstrated nasal polyps with dense eosinophilic infiltration and cultures grew polymicrobial bacteria. He underwent left eye evisceration, left lower eyelid ectropion repair, and second-stage sinus surgery. Postoperatively, there were improvements in pain, breathing, and smell. Nasal endoscopy displayed improved sinus patency, although with rapid polyp recurrence. Given concern for mucocele formation and potential impact on the right eye, bimonthly subcutaneous dupilumab was initiated. Following 5 doses, there was dramatic improvement, with symptom resolution, nasal bridge narrowing, sinonasal polyposis reduction, and fistulae closure (Fig. 1C). Subsequent CT showed dramatic polyposis involution and sinus clearing (Fig. 2, bottom). Severe chronic sinusitis with polyposis may exert mass effect on local structures including the brain, optic nerves, and globes. Among patients with severe, refractory, or recurrent polyposis, subcutaneous dupilumab may provide a dramatic clinical response.FIG. 1FIG. 2

Evaluation of gallbladder motility assessed by ultrasonography in dogs with hyperlipidemia.

The pathogenesis of gallbladder (GB) mucoceles in dogs is unknown. It has been proposed that hyperlipidemia could impair GB motility and contribute to GB mucocele formation. The objective of this study was to compare GB motility in dogs with hyperlipidemia to control dogs using ultrasonography. We hypothesized that hyperlipidemic dogs will have decreased GB motility compared with controls. Twenty-six hyperlipidemic and 28 healthy, age-matched control dogs were prospectively enrolled. Cholesterol and triglyceride concentrations were measured in all dogs. Hyperlipidemia was defined as hypercholesterolemia (>332 mg/dL) and/or hypertriglyceridemia (>143 mg/dL) using a biochemical analyzer. Ultrasound was performed before feeding, and 60 and 120 minutes after ingestion of a high fat diet. Gallbladder volumes (GBV) and ejection fractions (EF) were calculated. Hyperlipidemic dogs had significantly larger GBVs (ml/kg) before feeding and 60 minutes after feeding of 1.2 (0.4-7.5; P = .008) and 0.6 (0.1-7.2; P = .04) compared with controls 0.6 (0.2-2.6) and 0.4 (0.1-1.9), respectively. Severely hyperlipidemic dogs had significantly larger GBV at baseline, 60 minutes, and 120 minutes of 1.7 (0.6-7.5; P = .03), 1.3 (0.4-7.2; P = .02), and 1.3 (0.2-8.2; P = .04), respectively compared with mildly hyperlipidemic dogs. EFs at 60 and 120 minutes between controls, hyperlipidemic, and severely hyperlipidemic were all 0.3 at 60 minutes and 0.5, 0.3, and 0.3 at 120 minutes, respectively which were not statistically different. Hyperlipidemia leads to GB distention in dogs which could lead to retention of bile and gallbladder disease.

Large gallbladder Removed by Laparoscopic Cholecystectomy A Case report

Gallbladder stones impacted at neck of gallbladder can result in acute calculous cholecystitis with mucocele gallbladder. Laparoscopic cholecystectomy is performed in such cases as an emergency surgery. We present a case long gallbladder with mucocele formation that was managed by laparoscopic approach. The gallbladder removed was 27.8cm in length. This is the longest gallbladder removed by laparoscopic cholecystectomy in Pakistan so far. The patient had uneventful recovery.

Gallbladder microbiota in healthy dogs and dogs with mucocele formation.

To date studies have not investigated the culture-independent microbiome of bile from dogs, a species where aseptic collection of bile under ultrasound guidance is somewhat routine. Despite frequent collection of bile for culture-based diagnosis of bacterial cholecystitis, it is unknown whether bile from healthy dogs harbors uncultivable bacteria or a core microbiota. The answer to this question is critical to understanding the pathogenesis of biliary infection and as a baseline to exploration of other biliary diseases in dogs where uncultivable bacteria could play a pathogenic role. A pressing example of such a disease would be gallbladder mucocele formation in dogs. This prevalent and deadly condition is characterized by excessive secretion of abnormal mucus by the gallbladder epithelium that can eventually lead to rupture of the gallbladder or obstruction of bile flow. The cause of mucocele formation is unknown as is whether uncultivable, and therefore unrecognized, bacteria play any systematic role in pathogenesis. In this study we applied next-generation 16S rRNA gene sequencing to identify the culture-negative bacterial community of gallbladder bile from healthy dogs and gallbladder mucus from dogs with mucocele formation. Integral to our study was the use of 2 separate DNA isolations on each sample using different extraction methods and sequencing of negative control samples enabling recognition and curation of contaminating sequences. Microbiota findings were validated by simultaneous culture-based identification, cytological examination of bile, and fluorescence in-situ hybridization (FISH) performed on gallbladder mucosa. Using culture-dependent, cytological, FISH, and 16S rRNA sequencing approaches, results of our study do not support existence of a core microbiome in the bile of healthy dogs or gallbladder mucus from dogs with mucocele formation. Our findings further document how contaminating sequences can significantly contribute to the results of sequencing analysis when performed on samples with low bacterial biomass.

Gallbladder microbiota in healthy dogs and dogs with mucocele formation

Gallbladder microbiota in healthy dogs and dogs with mucocele formation

Appendiceal Mucocele Developed by Appendiceal Mucosal Prolapse: A Rare Case Report with Review of Literature

Appendiceal mucocele formation can be induced by mechanical obstruction due to various reasons; In very rare cases, mucosal prolapse of the appendix can also cause mucocele. We first present the case of a 38-year-old woman who was diagnosed with appendiceal mucocele associated with appendiceal mucosal prolapse. The lesion was first found by colonoscopy and treated surgically. On examining the specimen, it revealed a dilated lumen containing mucinous fluid at the proximal portion, a polypoid mass at the mid portion, and a slightly dilated distal portion filled with mucoid material. But there was no histologic evidence of polyp. We report the first case of mucosal prolapse of the appendix resulting in mucocele with a review of the concepts of prolapse.

Draf Type III Surgery for Delayed Formation of Lateral Frontal Sinus Mucocele with Repeated Inflammation Following Frontal Craniotomy: Case Report and Literature Review

Draf Type III Surgery for Delayed Formation of Lateral Frontal Sinus Mucocele with Repeated Inflammation Following Frontal Craniotomy: Case Report and Literature Review

Concha bullosa mucocele disguising as a nasal tumor in an epistaxis patient: A case report

Concha bullosa is the most common anatomical variant of middle turbinate in both children and adults. When the natural drainage of concha bullosa is obstructed, it leads to the formation of mucocele. It is confusing with nasal tumor when the entire nasal cavity gets occupied. Concha bullosa mucocele and profuse epistaxis is rarely found in the same patient. Both pathologies require appropriate management for the early recovery of the patient and prevent severe complications like concha bullosa mucopyocele from concha bullosa mucocele and hypovolemic shock from profuse epistaxis. We present a 74-year-old female who presented with profuse epistaxis from the left nasal cavity due to concha bullosa mucocele filling the right nasal cavity and pushing nasal septum towards left side which was confused with nasal tumor.

A multicenter retrospective study assessing progression of biliary sludge in dogs using ultrasonography.

BackgroundBiliary sludge (BS) frequently is identified on ultrasonographic examination and is described as incidental. It is hypothesized that biliary stasis and hypersecretion play a role in both BS and gallbladder mucocele (GBM) formation. Recent studies have documented similarities in composition of BS and GBM, and there are several examples of progression from BS to GBM in the veterinary literature.ObjectivesTo assess the relationship between the presence of BS and later development of GBM in dogs, over time periods >12 months.AnimalsA total of 154 dogs with BS and ultrasonographic follow‐up >12 months.MethodsMedical records were retrospectively collected from 9 UK‐based referral centers for all available time points. A semiobjective scoring system was used to track volume of BS within the gall bladder (GB) over time.ResultsTwenty dogs developed GBM during the study period. Shetland Sheepdogs (odds ratio [OR], 40.99; 95% confidence interval [CI], 3.61‐465.95; P = .003) and Border Terriers (OR, 11.66; 95% CI, 3.28‐46.63; P < .001) were independent risk factors for the development of GBM. Non–gravity‐dependent BS (NDBS) was noted to form before GBM development in 9/20 dogs, and breeds at‐risk for GBM were more likely to have NDBS. Odds for the development of GBM increased with BS score.Conclusions and Clinical ImportanceDogs with NDBS may be at risk for the development of GBM and a stratified BS scoring system could allow for semiobjective monitoring over time, particularly in at‐risk breeds.

Gastric Remnant Mucocele after Conversion of Vertical Band Gastroplasty to One Anastomosis Gastric Bypass: a Video Vignette.

Conversional bariatric surgery is a challenging procedure to patients as well as the surgeon. One anastomosis gastric bypass (OAGB) is a known safe conversional procedure after primary restrictive weight loss surgeries such as vertical banded gastroplasty (VBG). A very rare reported complication after these operations is the formation of mucocele of gastric remnant during pouch creation. This is a video report of diagnosis and management of gastric remnant mucocele after conversion of VBG to OAGB.

부비동 내시경 수술 후 발생한 전두동 점액농류 1예

Mucocele is a mucus-containing benign lesion, whose clinical characteristic of slow expansion may lead to erosion and remodeling of the bony sinus walls. When the internal content of the mucocele becomes infected, the lesion is called mucopyocele. The etiology of mucocele formation is multifactorial including inflammation, allergy, trauma, anatomical abnormality, previous surgery, fibrous dysplasia, osteoma, and ossifying fibroma. During the past decades since the introduction of endoscopic sinus surgery (ESS) for chronic rhinosinusitis (CRS), paranasal sinus mucoceles following ESS have been increasingly reported. However, giant mucopyoceles of the frontal sinus expanding cerebral parenchyma are extremely rare. In this case report, we describe a case of huge frontal sinus mucopyocele compressing left frontal lobe after ESS, which was successfully treated by wide marsupialization via the transnasal endoscopic approach. (J Clinical Otolaryngol 2021;32:276-280)

Point of Care Virtual Surgical Planning and Three-Dimensional Printing for Feminizing Foreheadplasty.

Point of Care Virtual Surgical Planning and Three-Dimensional Printing for Feminizing Foreheadplasty.