Orbitofrontal cholesterol granuloma masquerading as frontal sinus mucoceles: report of two cases.

BACKGROUND: Two cases of orbitofrontal cholesterol granuloma masquerading as frontal sinus mucoceles were reported to understand image findings, clinical and histopathologic features of orbitofrontal cholesterol granuloma to improve its diagnosis and treatment. CASE PRESENTATION: Two East Asian patients aged 41 and 27 without personal or familial medical or trauma history presented with the common complaint of proptosis and inferomedial displacement of the eyeballs. The computed tomography (CT) of both cases showed an irregularly shaped, well-defined lesion in the left frontal bone associated with bony erosion. The lesions resulted in the bone absorption of frontal bone and orbital roof, which extended into the superior orbital space. Anterior orbitotomy through subbrow incision by drainage and curettage resulted in a curative outcome. The histopathological examination revealed inflammatory granulation tissues, fibrous capsule wall, cholesterol clefts with altered blood pigments, and calcifications, consistent with the diagnosis of cholesterol granuloma. No recurrence was observed for one year after surgery in one case and three years in the other. CONCLUSIONS: When the following features are observed: orbital CT exhibits cystic lesion with irregular bone destruction in the superolateral orbit, magnetic resonance imaging (MRI) depicts lesions are hyperintense signals on T1 weighted images (T1WI), and T2 weighted images (T2WI), and the contrast-enhanced imaging reveals that the most of tumor is showed a non-significant enhancement, orbitofrontal cholesterol granuloma should be considered.

Compressive optic neuropathy due to posterior ethmoid mucocele.

Mucoceles are cystic formations characterized by the presence of mucus-secreting epithelial cells, which enlarge when the excretory duct becomes obstructed. Posterior ethmoidal mucoceles are rare conditions that can lead to severe ocular complications requiring immediate intervention. The close anatomical proximity of posterior ethmoidal mucoceles to the optic nerve underscores their significance. In this case report, we present a case of rapidly progressing compressive optic neuropathy secondary to a posterior ethmoidal mucocele. A previously healthy forty-six-year-old woman presented with sudden visual loss in her left eye, preceded by left-sided headache and periorbital pain. Clinical examination and imaging studies revealed an oval-shaped mass within the posterior ethmoid cell compressing the left optic nerve. Emergency surgery was performed to alleviate optic nerve compression, which successfully relieved periocular pain. However, the patient's visual acuity and visual field defect remained unchanged postoperatively. Thinning of the ganglion cell layer in the macula region was observed during follow-up examinations. The role of corticosteroids and antibiotics in visual rehabilitation and the impact of delayed surgical decompression on visual outcome remain subjects of debate. Additional cases of mucocele-associated optic neuropathy should be published and analyzed to establish optimal treatment approaches.

Isolated Orbital Apex Mucocele: A Case Study and Literature Review.

Isolated orbital mucocele without connection between the mass and paranasal sinuses is extremely rare and poorly understood. Literature review of these cases are very few and present more anteriorly in the orbit. Here, the authors present a 33-year-old female with an isolated left orbital apex mucocele without direct communication with the adjacent paranasal sinuses and other vital orbital structures. Endoscopic sinus surgery with marsupialization was performed, and an orbital mucocele was confirmed on histopathology. Although uncommon, previously reported cases, including our patient have remained disease free of recurrence for at least 1 year post-operatively.

Ophthalmic Presentation and Outcome for Sinonasal Mucoceles.

PURPOSE: The purpose of this study was to evaluate ophthalmic features and outcomes for patients who present with sinonasal mucoceles expanding into the orbit. PATIENTS AND METHODS: Retrospective chart review for patients seen in a specialist orbital clinic over 25 years, with a review of demographics, clinical characteristics, imaging features, and outcomes after treatment. RESULTS: Sixty patients (38 males; 63%) presented at a mean age of 51 years (range 3-89). Symptom duration was extremely variable (1 week-15 years) with a mean of 14 months and median of 4 months-the commonest being periorbital swelling (33/62 orbits) or ache (20 orbits), proptosis (30 orbits), and diplopia (19/50 patients without visual impairment; 38%). Of mucoceles affecting orbital function, 60/62 (97%) were of frontal and/or ethmoid sinus origin, and probable predispositions included past trauma (12/62 orbits) or prior ipsilateral sinus surgery (14 orbits). Forty-two of the 59 (71%) patients who underwent sinus surgery had complete resolution of symptoms within 6 months. Of 10 orbits presenting with moderate to severe visual loss (Snellen 20/60 or worse), the acuity improved in 7/10 (70%) of these after sinus surgery. Although 12/62 (19%) of eyes presented with epiphora, this persisted after sinus surgery in 9 orbits, and areas of occult malignant change were found in 3/9 (33%). CONCLUSION: Sinus mucoceles expanding into the orbit can cause significant globe displacement, dysmotility, or visual impairment. Symptoms usually resolve within several months after functional sinus surgery, but where symptoms persist (particularly periorbital swelling or epiphora) this might indicate underlying secondary causes, such as occult malignancy.

Bilateral Rhinogenous Optic Neuropathy Caused by the Regrowth of a Large Sphenoid Sinus Mucocele.

A 59-year-old man, who had a history of left blind at 36 years old, suddenly lost right visual acuity. Magnetic resonance imaging revealed a large left sphenoid sinus cyst, which protruded intracranially. The cyst was fenestrated by endoscopic sinus surgery, but his right vision did not recover. Ten cases of bilateral rhinogenous optic neuropathy caused by mucocele have been reported, and the cause was sphenoid sinus in 9 cases. Postoperative visual acuity in these cases was poor, especially in slow progressive cases, because it was diagnosed as an unknown cause, and surgery was delayed. Rhinogenous optic neuropathy caused by mucocele should be differentiated from bilateral visual impairment of unknown cause. The authors highlight the importance of early diagnosis of sphenoid sinus mucocele and fully informing patients about the future risk of bilateral visual impairment, even if they are asymptomatic or have been treated.

The effect of flushing of the common bile duct on hepatobiliary markers and short-term outcomes in dogs undergoing cholecystectomy for the management of gall bladder mucocele: A randomized controlled prospective study.

OBJECTIVE: To determine the effect of flushing of the common bile duct (CBD) on hepatobiliary markers and short-term outcome in dogs undergoing cholecystectomy for the management of gallbladder mucocele (GBM). STUDY DESIGN: Randomized, controlled, prospective study. ANIMALS: Thirty-two client-owned dogs. METHODS: Dogs were allocated randomly to either a "flush" group or a "non-flush group." Flushing was performed in a normograde fashion, followed by a routine cholecystectomy. Data collected included presenting clinical signs, preoperative and 3-day postoperative hepatobiliary markers (alkaline phosphatase, ALP; alanine aminotransferase, ALT; gamma glumatyl-transferase, GGT; bilirubin; cholesterol; triglycerides), duration of hospitalization, and complications. These data were compared between groups. RESULTS: Sixteen dogs were enrolled in each group. One dog (in the flush group) was excluded following diagnosis of hepatic lymphoma. Border terriers were overrepresented (20/31). Overall, there were marked reductions from preoperative to 3 days postoperative in serum bilirubin (p = .004), ALP (p = .020), ALT (p < .001), GGT (p = .025), and cholesterol (p < .001) values. There was no difference in any marker between groups. Survival to discharge was 90.3% (28/31 dogs). CONCLUSION: Cholestatic markers decreased significantly 3 days postcholecystectomy. No short-term clinical or clinico-pathological benefits were identified when flushing the CBD in dogs undergoing cholecystectomy for GBM. CLINICAL SIGNIFICANCE: The findings of the study do not support routine flushing of the CBD during cholecystectomy for GBM in dogs.

Management of mucocele of the appendix with peritoneal dissemination in pregnant women: a case report and literature review.

BACKGROUND: This is the first case report of a patient with a twin pregnancy who developed appendiceal mucocele (AM) with peritoneal dissemination in the context of endometriosis and ovarian hyperstimulation. A review of the literature on AM during pregnancy, with a focus on management, is provided as well. CASE PRESENTATION: A 36-year-old woman was admitted to the emergency department for abdominal pain in the right iliac fossa at 17 weeks of amenorrhoea (WA). She was pregnant with twins, achieved by in vitro fertilisation (IVF) and complicated by ovarian hyperstimulation syndrome. Abdominal ultrasonography indicated a diagnosis of uncomplicated acute appendicitis. However, the histopathological findings revealed low-grade appendicular mucinous neoplasia (LAMN) with peritoneal dissemination of acellular mucin. Maximal cytoreductive surgery with hyperthermic intraperitoneal mitomycin chemotherapy was performed post-partum. CONCLUSIONS: Specialised management is required for AM during pregnancy and must take into account the progress of the pregnancy, the histology of the AM and the risk of perforation.

Tongue Mucoceles: a retrospective clinic-pathological evaluation of 240 cases.

BACKGROUND: Minor salivary glands can be found in the ventral and anterior part of the tongue; these glands can rarely develop mucoceles that, due to their rarity and their unusual clinical appearance, may present an interesting differential diagnosis. Mucoceles appear as an exophytic, sometimes pedunculated, lesion, which is a feature that is due to the absence of a capsule; thus, the glands are right beneath the mucosa and over the muscle tissue. The aim of this article is to retrospectively present and discuss the anatomy, pathology, clinical features and therapy of several cases of Blandin-Nunh mucoceles collected from two different institutions. METHODS: A retrospective case review was carried out in two university institutions, retrieving all cases of tongue mucoceles from 1999 to today. Two oral pathologists reviewed all the slides, confirming the diagnosis. Demographic data of the patient, anatomic location and clinical appearance were retrieved from clinical charts, together with the type of surgical procedure and possible relapses. RESULTS: A total of 240 cases of tongue mucoceles were gathered from the archives: the mean age was 22 years (DS = 14,7; Range 2-83), 126 were females (52,5%, mean age 22,7 years, DS = 16,5; Range 2-83), and 114 were males (47,5%, mean age 20,9 years, DS = 12,4; Range 3-73); in all cases, a history of trauma was reported. The ventral surface was the most frequent location (224 cases - 93,3%), and in the great majority (235 cases - 97,9%), pathology revealed mucous spillage with a wall formed by fibrous connective and granulation tissue with no epithelium lining the cavity. Superficial mucocele and sclerosing sialoadenitis were the more frequent pathological variants (21 cases - 8,8%). All lesions were treated with excision and enucleation of the servicing gland. The healing was uneventful in all cases, but there were four recurrences and two cases of sensory paraesthesia of the border of the tongue, all in males, except one case of paraesthesia in a female. CONCLUSIONS: Tongue mucoceles must be differentiated from many benign and malignant lesions. For this reason, surgical removal of the lesion and of the associated gland with a pathological exam is mandatory. In fact, the anatomical location of the glands and the possible pathological variants must be considered to reach a correct diagnosis and diminish possible relapses. TRIAL REGISTRATION: CE-Muc_Ton_3/2023.

Management of an extensive frontal pyocoele using an external approach: A case report.

Pyocoeles of the paranasal sinuses are pus-filled cavities seen in the sinuses and develop from the infection of the mucocoeles. Pyocoeles most commonly form in the frontal sinus. Endoscopic Sinus Surgery is currently the mainstay of the treatment. We present the case of an 85-year-old female who presented to the ENT OPD of Jinnah Hospital/AIMC, Lahore, in October 2017 with complaints of progressive symptoms of intractable left frontal pain, double vision, and an associated swelling below the medial half of the left eyebrow. A CT scan was done and a diagnosis of extensive frontal pyocoele was made. She was treated successfully with an external approach and remained without any complications till 13-month follow-up after surgery; exceptional cosmetic results were achieved. We wish to bring attention to the possibility of an external approach being used as a primary intervention, if deemed appropriate.

Paranasal mucoceles in children with cystic fibrosis: Management of a not so rare clinical condition.

PURPOSE: Paranasal mucocele (PM) is reported as a complication in children with cystic fibrosis (CF) in up to 4% of patients. The objective of this study was to identify PMs in the personal large series of children with CF and to assess their diagnosis and treatment. MATERIAL AND METHODS: Medical records of children with CF and PM who were admitted and treated by means of endoscopic nasal surgery between 2004 and 2020 were retrospectively reviewed. RESULTS: Thirty-four patients were included in the study (mean age 7.7 years). CT scan of sinuses showed a total of 53 PMs. Nasal endoscopic findings suggestive for PM were present in almost 80% of patients. PMs were located in the maxillary, ethmoid, and sphenoid sinuses in 29/34 (85.3%, bilateral in 17 cases), 4/34 (11.8%) and 1/34 (2.9%) patients, respectively. Marsupialization of PMs was performed in all patients using an endoscopic transnasal approach. No complications were observed. Resolution of symptoms and normalization of the endoscopic nasal picture was evident in all patients. After a mean follow-up of 85 months, no recurrences were observed. CONCLUSIONS: To the best of our knowledge, this is the largest series of CF patients with PMs. Even if not frequently reported in the literature, PMs should not be considered an uncommon finding in patients affected by CF. Routine nasal endoscopy is mandatory to favor early diagnosis. Endoscopic transnasal marsupialization represents the gold standard of care for patients with CF and PM(s).

Orbital and Eyelid Inflammation With "Muciphages" and Extravasated Mucin From an Ethmoido-orbital Mucocele.

A 22-year-old woman presented with an acute compressive optic neuropathy due to a ruptured ethmoido-orbital mucocele. She underwent urgent orbital decompression and drainage of the mucocele via an endoscopic approach. Postoperatively, her course was complicated by an orbital compartment syndrome supervened, exhibiting severe eyelid edema caused by infiltration with mucin and mucin-containing macrophages ("muciphages"). Biopsy of the eyelid showed infiltration with "muciphages," macrophages laden with extravasated mucinous material. This is the first report that documents the clinical and histopathologic course of orbital inflammation following mucocele extravasation into the orbit and eyelids.

Bilateral Ethmoidal Mucocele Causing Proptosis in a Child.

OBJECTIVES: Paranasal sinus mucocele is a benign cystic lesion, lined with respiratory epithelium and filled with mucoid secretions, which is generally seen in the ethmoid or frontal sinuses. Inflammation, trauma, fibrosis, neoplasm, and previous surgery play a role in etiology. Treatment of this condition requires marsupialization and drainage. Endoscopic surgery is the gold standard procedure. METHODS: A 2-year-old boy was referred to our clinic with a diagnosis of bilateral ethmoid mucocele causing proptosis in the right eye. General clinical examinations and routine blood analysis revealed normal health condition of the patient and comorbidities such as cystic fibrosis (CF) and/or ciliary dysfunction were excluded. The patient was operated with an endoscopic endonasal approach. RESULTS: As far as we know, our article reports the first case of bilateral ethmoidal sinus mucocele with no underlying comorbidity, such as cystic fibrosis or ciliary dyskinesia. CONCLUSIONS: Children are rarely affected by mucocele and if present at early age, there is usually an underlying cause. In pediatric patients with signs and symptoms suggestive of a mucocele, it is important to rule out other benign and malignant paranasal sinus pathologies. Marsupialization and drainage via endoscopic endonasal approach is the preferred treatment modality.

The anatomical basis and rational for the transoral approach during the surgical excision of the sublingual salivary gland for the management of plunging ranula.

PURPOSE: The aim of this study was to illustrate and confirm that the complete intraoral excision of the sublingual gland alone, is anatomically the most rational approach, for the management of plunging ranula. MATERIAL AND METHODS: We retrospectively reviewed clinical charts of diagnosed HIV-infected patients presenting with plunging ranula. The intraoral approach was used for the complete excision of the sublingual gland with evacuation of the pseudocystic content only. Neither extra oral approach, nor ranula dissection / drainage was performed. The surgical procedure was performed using local anesthesia. Pre- and postoperative MRI-scan investigations were recorded. Histological reports were documented to confirm the diagnosis of oral mucocele. Patients were clinically monitored. RESULTS: We identified 90 adults presenting with oral mucocele, type ranula. Seventy (77%) of them were diagnosed with HIV infection. Plunging ranula was recorded in 35 (50%) patients from the latter group. The study enrolled 11 operated patients whose files contained useable data, including an acceptable follow-up period. The postoperative follow-up period ranged from three to 15 months. The clinical and postoperative MRI-scans of operated patients demonstrated satisfactory results. There were neither postoperative complications nor recurrence of ranula reported. CONCLUSION: The location of the sublingual gland in the floor of the mouth coupled with the physio-pathogenesis of the plunging ranula, makes the transoral complete excision of the offending gland, with the intraoral evacuation of the pseudocyst, anatomically the most rational approach for plunging ranula management. There is no need for cervical approach, ranula dissection and/or postoperative placement of drainage.

Hypohidrotic ectodermal dysplasia: a case report.

Hypohidrotic ectodermal dysplasia is a common variation of ectodermal dysplasia, characterized by hypohidrosis (or anhidrosis), hypotrichosis, hypodontia, and other distinct facial features. Furthermore, ocular tissues of ectodermal origin may also be affected in this disease. The most common ocular manifestations of hypohidrotic ectodermal dysplasia are dry eye, madarosis, alterations in the meibomian glands, abnormalities in the nasolacrimal duct, and infantile glaucoma. Herein, author reports a case of hypohidrotic ectodermal dysplasia in a 12-year-old Indian boy with dry eye and lacrimal sac mucocele.

[A rare case of appendiceal mucinous tumor with myxoglobulosis]. / Un cas rare de tumeur mucineuse appendiculaire avec myxoglobulose.

Myxoglobulosis is a rare macroscopic form (<1%) of appendiceal mucocele characterized by opaque mucin beads. This entity, still unknown in clinical practice, can only be confirmed by anatomopathological examination. Many histological diagnoses that may impact the prognosis of patients should be discussed in the presence of myxoglobulosis, including neoplastic causes. We report the rare case of myxoglobulosis, whose histopathological management concluded the diagnosis of low-grade appendicular mucinous neoplasm.

Dermoscopy of oral mucocele: three types of extravasation mucoceles

Background/aim: Dermoscopy is a diagnostic tool that assists in imaging the epidermis and dermis. Although it has also started to be used to diagnose nonmelanocytic lesions recently, it has not been tested much on oral mucosal masses, such as oral mucoceles. This study aimed to investigate whether dermoscopy is a valuable tool in diagnosing oral mucoceles. Materials and methods: In this study, the clinical and dermoscopic features of 21 oral mucocele lesions of 21 patients (11 females, 10 males) aged between 6 and 38 years who were confirmed histopathologically were evaluated. Results: Of the lesions studied, 95.2% (20) were extravasation and 4.8% (1) were retention mucoceles. The nonvascular structures were determined as white areas (61.9%, 13), erythema (57.1%, 12), purplish-gray background (52.3%, 11), ulcer (30%, 8), yellowish- orange areas (23.8%, 5), crust (14.2%, 3), starburst pattern (0.95%, 2), and bleeding (0.47%, 1). Dermoscopically, 40% of extravasation mucoceles were classified as type 1 (8 patients), 25% as type 2 (5 patients), and 35% as type 3 (7 patients). Conclusion: We concluded that there are 3 types of extravasation mucoceles dermoscopically and clinically, and these types may be stages of transition between each other.

Clinical Analysis of Sphenoid Sinus Mucocele With Initial Neurological Symptoms.

BACKGROUND AND OBJECTIVES: Neurological manifestations associated with sphenoid sinus mucocele (SSM) are easily misdiagnosed due to nonspecific symptoms. The objective is to analyze and report the clinical features of SSM presenting with neurological manifestations, to allow an earlier diagnosis and more timely intervention for this disease. METHODS: This was a retrospective cross-sectional study including 19 patients. The detailed clinical information of 19 patients with the initial symptom of neurological manifestations caused by SSM presenting at the Second Affiliated Hospital of Wenzhou Medical University between January 2000 and May 2018 were retrospectively analyzed. Collected data including symptoms, signs, neuroimaging, and pathologic diagnoses. RESULTS: There were eleven males and 8 females, and their ages ranged from 23 to 71 years. Headache was the most frequent symptom, in 12 of the 19 patients presenting as the initial symptom. The visual disturbance included visual loss (4/19), diplopia (3/19), and another patient had both visual loss and diplopia. Neurophysical examination found that 4 patients presented with oculomotor nerve palsy, 4 patients had optic nerve or abducens nerve palsy, and 1 patient had optic neuropathy, oculomotor nerve palsy and abducens nerve palsy simultaneously. All patients underwent endoscopic surgery and had postoperative clinical symptom improvement. CONCLUSIONS: Headache is the most common symptom of SSM and should be on the differential diagnosis of patients presenting with headache, even if in isolation. The results suggest that CT and MRI are the best tools in diagnosis of SSM and endoscopic sphenoidotomy is a safe and effective method in the treatment of SSM.

Identification of predictive variables for the recurrence of oral mucocele.

BACKGROUND: Oral mucocele is the most common minor salivary gland lesion with good prognosis after surgical removal. However, its recurrence is not rare, sometimes bothersome. This study aimed to identify the possible predictive variables affecting the recurrence rate of oral mucocele. MATERIAL AND METHODS: The histoclinical data of 164 patients diagnosed with oral mucocele were retrospectively obtained by reviewing dental records. The predictive variables for its recurrence were identified by analyzing its recurrence rate according to clinical variables. RESULTS: The recurrence rate showed the significant differences according to location and age. Oral mucocele recurred with significantly higher frequency on the ventral mucosa of tongue (50.0%) than on the labial/buccal mucosa (8.8%). Its recurrence was significantly more common in the younger patients (aged < 30 years, 16.0%) than in the older patients (aged > 30 years, 4.4%). However, there was no significant difference in recurrence rates between surgical procedures using scalpels and those using lasers. CONCLUSIONS: Patients with oral mucocele should be more carefully informed of its possible recurrence, especially when it is found on the ventral surface of the tongue or in a younger population.