ABSTRACTBehçet's disease is a chronic, relapsing multisystem vasculitis known for its highly variable clinical spectrum. We report the case of an 18‐year‐old male who presented with progressive headache and facial swelling. Imaging revealed extensive thrombosis involving the dural venous sinuses, superior vena cava, and left brachiocephalic vein. Despite the absence of ocular involvement or genital ulcers, a diagnosis of Behçet's disease was established based on recurrent oral ulcers, cutaneous findings, vascular thrombosis, and a positive pathergy test, fulfilling the International Criteria for Behçet's Disease. The patient was treated with corticosteroids, azathioprine, colchicine, and rivaroxaban, resulting in clinical and radiologic improvement. This case highlights a rare and aggressive vascular manifestation of Behçet's disease in an adolescent, emphasizing the need for early diagnosis and immunosuppressive management to prevent long‐term complications.

Background: Periapical surgery is indicated for persistent periapical lesions that do not respond to conventional endodontic therapy, yet postoperative recovery is often hindered by pain, swelling, and delayed healing. Platelet-rich fibrin (PRF) membranes are autologous biomaterials with regenerative potential, capable of modulating inflammation and promoting tissue repair. Methods: This preliminary randomized controlled trial evaluated the effectiveness of PRF membranes in improving postoperative outcomes—specifically pain, swelling, and quality of life—after apicoectomy. Twenty patients requiring periapical surgery were randomly allocated to a PRF group (n = 10) or a control group (n = 10). In the PRF group, autologous PRF membranes were applied over the resected root-end and into the osteotomy cavity before flap closure. In the control group, no PRF membranes or any additional biomaterial were applied, apart from the standard root-end filling material (MTA), which was identically used in both groups as part of the routine apicoectomy protocol. All patients were blinded to allocation, and outcomes were assessed by an independent blinded evaluator. Facial swelling was quantified by 3D facial scanning, pain was recorded daily using a visual analog scale (VAS), and quality of life was evaluated with the PROMIS-29+2 Profile. Results: The PRF group showed significantly reduced swelling (mean volume difference, 7.12 cm3; p = 0.025), lower pain scores (VAS: 1.80 ± 1.22 vs. 3.80 ± 2.44; p = 0.034), and improved quality-of-life domains, including higher Physical Function (p = 0.032) and lower Sleep Disturbance (p = 0.008) scores. Conclusions: Within the limitations of this pilot study, PRF membranes enhanced postoperative recovery after periapical surgery by reducing swelling and pain while improving patient-reported outcomes. Larger multicenter trials are needed to confirm these preliminary findings.

Hereditary angioedema (HAE) affects 1.22 people per 100,000 worldwide, and can cause fatal airway obstruction due to laryngeal oedema. Diagnosis is often delayed by several years, due to rarity, and similarity to more common histamine-mediated angioedema. Approximately 20-25% of cases have a de novo genetic variant, and no family history. Clinically, HAE is characterised by recurrent, non-pruritic swelling of the skin and mucosal tissues, without urticaria, which does not respond to antihistamines, corticosteroids or adrenaline. Definitive diagnosis requires demonstration of reduced C1inhibitor levels or function, which cannot be provided on an emergency basis. Complement C4 levels are always reduced during acute attacks, and awareness of this surrogate test in an appropriate clinical setting allows appropriate management of previously undiagnosed patients, in emergency settings. A female in her thirties presented to the emergency department (ED) with a 36-hour history of progressive facial swelling, including lip, tongue and uvular oedema, with concerns for airway compromise, requiring ICU admission for airway monitoring. She reported three similar previous episodes, and exhibited no urticaria or features of an allergic reaction. Treatments for histaminergic angioedema were ineffective. Given slowly-evolving episodes of angioedema, absence of urticaria or response to therapy, HAE was suspected. Urgent complement testing showed a markedly reduced C4 level with a result available in 1h. Based on the patient's presentation and very low C4, a presumptive diagnosis of an acute HAE attack was made, and treatment with Icatibant was given. The patient responded rapidly to treatment, with full resolution of symptoms. Subsequent testing confirmed a diagnosis of HAE with C1 esterase inhibitor deficiency. This case highlights the importance of considering HAE in patients presenting with unexplained slowly-evolving angioedema without urticaria, when traditional histamine-directed therapies fail, especially when airway compromise is imminent. Awareness that C4 levels are universally low during HAE attacks, is key. C4 testing is widely available and prompt communication with biochemistry or immunology laboratories facilitates rapid turnaround. Urgent presumptive diagnosis facilitates appropriate emergency management and prompt referral for definitive diagnosis and management. Early identification and management of HAE can significantly reduce morbidity and mortality associated with this condition.

An uncommon, histologically benign, but aggressive, tumor of the jaws, ameloblastoma develops from odontogenic epithelium and has the capacity to extensively destroy jaw bones and infiltrate surrounding tissues. After incomplete treatment, recurrences are frequent. The orbit, anterior cerebral cavity, temporal and infratemporal fossa, and other challenging locations can experience recurrences. We report a case of recurrent complex cystic ameloblastoma over the right side of the face in a 31-year-old gentleman who presented with right facial swelling, serous discharge, intermittent pain, and decreased mouth opening for 2 years. Computed tomography (CT) head and neck with 3D reconstruction showed a complex solid-cystic multiloculated mass lesion in the right infratemporal fossa with large cystic components, septations, solid enhancing components, and peripheral calcifications, closely abutting the parotid gland, pterygoids, mandible, and maxillary antrum. Needful evaluation done. The patient underwent wide local excision of the tumor, right infrastructure maxillectomy, superficial parotidectomy, modified radical neck dissection, and microvascular alanine aminotransferase (ALT) free flap reconstruction. Complete surgical resection with negative margins is the hallmark for curative resection. Local recurrences are rare and usually amenable to re-resection. Postoperative radiotherapy for patients at increased risk of local recurrence improves local tumor control.

ABSTRACTNecrotizing fasciitis (NF) is a rare but serious life‐threatening condition in children that can be easily overlooked due to its atypical presentation and absence of common risk factors. This case involves a 10‐year‐old girl with a history of non‐transfusion‐dependent thalassemia and epilepsy who developed cervicofacial NF without any prior trauma or infection. The case highlights the importance of early recognition and prompt surgical intervention. In patients with underlying conditions, the rapid progression of NF underscores the need for a high index of suspicion when faced with unusual facial swelling or skin changes. Early surgical debridement and appropriate antibiotics are critical to improving outcomes and minimizing complications in pediatric NF cases.

ObjectiveThis study aimed to evaluate the clinical efficacy of intraoral vacuum-assisted drainage (VAD) in reducing postoperative inflammatory complications—pain, swelling, and trismus—following the surgical extraction of impacted mandibular third molars (M3M).Materials and methodsA prospective, randomized, triple-blinded, sham-controlled clinical trial was conducted with 36 systemically healthy participants undergoing surgical removal of a single M3M. Participants were randomly assigned to either the VAD group (n = 18), receiving three sessions of intraoral negative pressure therapy via a custom-made splint, or the sham-control group (n = 18), in which the apparatus was applied without suction. Primary outcomes included pain intensity, facial swelling, and maximum mouth opening (MMO), measured preoperatively and on the 2nd and 7th postoperative days. Secondary outcomes were NSAID consumption and oral health–related quality of life (OHIP-14). Statistical analysis was conducted with a significance level of 0.05.ResultsThe VAD group showed significantly lower pain scores at all postoperative time points compared to controls (p < 0.05). Swelling was significantly reduced on postoperative day 2 (p = 0.019), and MMO was significantly greater on both postoperative days 2 and 7 (p < 0.001 and p = 0.001, respectively). Nonsteroidal anti-inflammatory drug consumption was significantly lower in the VAD group (8.1 ± 3.2 vs. 12.8 ± 4.6 tablets, p = 0.001), and OHIP-14 scores indicated improved quality of life on postoperative day 7 (p = 0.021).ConclusionsWithin the limitations of this preliminary trial, vacuum-assisted drainage showed potential in reducing early postoperative complications following M3M surgery. These findings support further investigation into its clinical application.Clinical relevanceThis study introduces an innovative intraoral vacuum-assisted drainage technique that enhances patient recovery after mandibular third molar surgery. By reducing inflammatory complications and analgesic dependency, it offers a promising adjunct to standard postoperative care, particularly in patients where NSAID use is limited or adverse effects are a concern.Trial registrationClinicalTrials.gov Identifier NCT06758258. Date of Registration 26/11/2024 (retrospectively registered).

Background To optimize the postoperative cold compress protocol for mandibular impacted third molar extraction. Methods Subjects were randomly divided into two major groups. The first group compared continuous vs. intermittent cold compress application, while the second group evaluated the duration of cold compress therapy. Postoperative outcomes, including pain intensity, facial swelling, trismus, and wound hemorrhage, were systematically analyzed. Results Continuous cold compress application within the first 6 h postoperatively demonstrated superior efficacy over intermittent application in alleviating pain, reducing swelling, improving mouth opening, and minimizing hemorrhage incidence. Cold compress application during postoperative D1 significantly controlled hemorrhage and mitigated acute pain. Prolonged therapy to D3 further enhanced facial edema reduction and trismus resolution. Conclusions A protocol of continuous cold compress application for 6 h daily during the initial 3 postoperative days significantly reduces complications, offering optimal clinical outcomes.

Surgical extraction of third molars is a common oral and maxillo-facial surgery procedure frequently associated with postoperative complications, such as pain, swelling, and trismus. Photobiomodulation (PBM), also known as Low-Level Laser Therapy (LLLT), involves the use of low-intensity laser to promote tissue healing, reduce inflammation, and relieve pain. This study evaluated the effectiveness of PBM following surgical extraction of mandibular third molars. This was a prospective, multicenter, randomized, triple-blind clinical trial conducted across five Italian centers. Seventy-nine patients were randomly assigned to a test group (PBM) or control group (no PBM). PBM was delivered immediately after surgery and on the following two days, using a multiband diode laser (445, 660, and 970nm). Primary outcomes were postoperative pain, facial swelling, and trismus. Secondary outcomes included health-related quality of life and analgesic consumption. Covariates included patient age, gender, the extracted tooth and the impaction classification according to Pell & Gregory and Winter. The time of the surgical procedure, measured in seconds from incision to final suture, was also documented. Descriptive statistics were calculated for all variables. Normality was assessed using the Shapiro-Wilk test, and homogeneity of variances was evaluated using Levene's test. Between-group comparisons for continuous outcomes were performed using Student's t-test or the Mann-Whitney U test, depending on data distribution. Categorical variables were analyzed with chi-square or Fisher's exact test. A p-value< 0.05 was considered statistically significant. No statistically significant differences were found between groups for any of the measured outcomes. However, trends favored the PBM group, particularly regarding reduced pain and improved quality of life. The study was powered to detect a large effect size (Cohen's d=0.8); therefore, the lack of statistical significance suggests that any true effect, if present, is likely smaller than this threshold. Further studies with larger sample sizes and standardized protocols are needed to explore smaller yet clinically relevant effects.

Four children were treated at the Ophthalmology Department of Beijing Children's Hospital affiliated with Capital Medical University, including a female patient aged 9 years and 11 months with redness and swelling of the forehead accompanied by bilateral eyelid swelling for 3 days, a male 14-day-old infant with progressive worsening of the right eye redness and swelling accompanied by high fever, nasal congestion and a runny nose for 7 days, a boy aged 5 years and 9 months with the left eye and facial redness and swelling, black skin deposition, rupture, pus discharge and high fever for 2 days, and a 7-year-old male with the right eyelid redness and swelling accompanied by fever and acute lymphoblastic leukemia for 2 hours. The comprehensive blood routine examination showed C-reactive protein levels exceeded 140 mg/L and white blood cell counts exceeded 20×109/L in all patients. Bacterial cultures of ocular secretions or pus were mainly positive for Streptococcus pyogenes, Acinetobacter baumannii, methicillin-resistant Staphylococcus aureus, and Pseudomonas aeruginosa. Orbital imaging revealed orbital abscess formation (Chandler stage 4) in 2 patients and subperiosteal abscess and necrotizing fasciitis (Chandler stage 3) in 2 patients. According to the scoring criteria for the systemic inflammatory response syndrome, all four cases were diagnosed with severe orbital cellulitis. Systemic and ocular antibiotics were administered, along with local debridement and abscess incision and drainage. Four children recovered without any recurrence of infection. Two of them developed late-onset inner canthal deformity and eyelid abnormality, which were resolved by corrective surgery.

Abstract OBJECTIVE To synthesize existing literature on JPOF and present a detailed case of a 22-year-old woman with maxillary, frontal, and ethmoidal sinus involvement, highlighting diagnostic features, surgical management, and short-term outcomes. METHODS A review of articles on JPOF was conducted, focusing on clinical presentation, radiologic findings, histopathology, and treatment strategies. Concurrently, we analyzed the case of a 22-year-old female who presented with progressive facial swelling and nasal obstruction. Diagnostic work-up included computed tomography (CT) and magnetic resonance imaging (MRI), followed by histopathological confirmation via endoscopic biopsy. The patient underwent endoscopic-assisted excision with intraoperative navigation to ensure complete lesion removal while preserving adjacent structures. RESULTS Imaging revealed a well-demarcated, expansile lesion with internal calcifications. Histology demonstrated psammoma-like bodies and fibroblastic proliferation, confirming JPOF. Surgical excision achieved clear margins without intraoperative complications. At three months postoperatively, the patient remained symptom-free with no radiologic evidence of recurrence. Literature review underscores the importance of early diagnosis, meticulous surgical planning, and the role of advanced imaging in minimizing morbidity. CONCLUSION JPOF demands a high index of suspicion, accurate imaging, and histopathology for diagnosis. Endoscopic-assisted resection with navigation yields favorable short-term outcomes. Given the lesion’s recurrence risk, long-term surveillance and further research into its molecular pathogenesis are essential to optimize management.

ABSTRACTPlasmablastic lymphoma (PBL), a rare aggressive B‐cell malignancy linked to HIV, is increasingly noted in extraoral sites beyond its traditional oral cavity presentation. This case report details a 48‐year‐old HIV‐positive woman with prior cerebral toxoplasmosis, presenting with facial swelling, pain, proptosis, and dysphagia despite antiretroviral therapy and cotrimoxazole prophylaxis (CD4 120 cells/μL, suboptimal viral suppression). Imaging identified a sinonasal mass with nasopharyngeal involvement and lymphadenopathy, with immunohistochemistry confirming PBL (MUM1+, weakly CD79a+, CD20‐, CD138‐, EBER‐). Treatment comprised six cycles of dose‐adjusted EPOCH with bortezomib (1.3 mg/m2 on days 1, 8, 15), intrathecal prophylaxis, and supportive care for recurrent neutropenia, achieving a complete metabolic response on interim FDG‐PET/CT after three cycles, with follow‐up CT showing pansinusitis. The patient is now eligible for autologous stem cell transplantation and consolidative radiotherapy, highlighting the diagnostic complexity of CD138‐negative extraoral PBL and the efficacy of bortezomib‐augmented EPOCH in this high‐risk case.

Introduction: Dexamethasone is widely used to minimize postoperative pain, swelling, and trismus following third molar surgery; however, the optimal route of administration remains debated. Objective: To compare the clinical effectiveness of submucosal, intramuscular, and intravenous administration of dexamethasone in reducing postoperative complications after mandibular third molar extraction. Methods: This randomized controlled study was conducted at a private hospital in Multan from January to June 2024 after obtaining IRB approval. Thirty patients indicated for surgical removal of impacted mandibular third molars were randomly assigned to two groups (n = 15). Group A received 8 mg dexamethasone via the submucosal route at the surgical site, while Group B received the same dose through the systemic route (intramuscular or intravenous). Postoperative pain (VAS), facial swelling (linear measurement), and mouth opening (interincisal distance) were evaluated at 24 hours, 48 hours, and 7 days postoperatively. Data were analyzed using SPSS 26 with significance set at p &lt; 0.05. Results: The mean age of participants was 26.8 ± 5.7 years, with an equal gender distribution. Both groups showed statistically significant reductions in postoperative pain, swelling, and trismus (p &lt; 0.05). The submucosal group exhibited superior outcomes in pain and facial swelling reduction, particularly within the first 48 hours, though differences were not statistically significant. Conclusion: All routes of dexamethasone administration effectively reduced postoperative discomfort following third molar surgery. The submucosal route provided a slight clinical advantage, likely due to its localized anti-inflammatory effect, making it a practical and minimally invasive alternative for routine use.

Does Injection of Dexamethasone Into the Pterygopalatine Fossa Reduce the Side Effects of Third Molar Surgery?

Utility of diagnostic sinus CT for diagnosis of invasive fungal sinusitis in immunocompromised children.

Abstract Introduction A previously fit-and-well 12-year-old boy, who was born and raised in the UK, traveled with his family to Islamabad, Pakistan over the Christmas holidays. Ten days into their vacation, he and his mother developed watery diarrhoea. While his mother’s symptoms self-resolved within five days, the patient developed worsening watery stools which were eventually associated with fresh red blood. Following their return to the UK, he developed a fever and red, watery eyes. He was brought to A/E and the patient admitted to having pain and burning sensation when passing urine as well. He also occasionally vomited for two days. Case description In hospital, he was started on IV fluids and IV ceftriaxone. He was prescribed regular ondansetron. Blood tests were sent which showed raised white cell count (14.6 à 33.3), raised neutrophils (9.8 à 29.3) and raised inflammatory markers (CRP 51 à 237 à 314). Eye swabs were negative. Stool samples were sent for microscopy and culture upon admission on day 11 of symptoms, but failed to grow any organisms on culture. His stool PCR upon admission however, tested positive for: Shigella toxin + astrovirus. On day 15 of illness, he woke up with bilateral, painful swelling of the face as well as pain and swelling in both ankles. Moreover, he complained of pain in his left big toe, which had become red and swollen. He was started on regular paracetamol and ibuprofen as per the rheumatology team’s advice, but in view of the facial swelling worsening and not settling with regular pain relief, he had input from the pain management team. Oral morphine was therefore started. An ultrasound of the parotid gland was arranged by the ENT team. Following discussion with the microbiology team, and after three days of IV ceftriaxone, he was switched to a five day course of IV azithromycin. On day 17 of illness, and in view of the worsening swelling of both parotid glands, he received a three day course of IV hydrocortisone as per advise from the ENT team. Following this, the parotid gland swelling reduced in size. Additionally, he was started on IV tazocin alongside his five-day course of azithromycin. His vomiting stopped whilst on regular ondansetron. Discussion By day 20 of illness, he was reviewed by the rheumatology team and the IV hydrocortisone was stopped. Immunology bloods were sent. His diarrhoea was persistent with a minimum of three loose bowel motions per day. His pain was managed well on paracetamol, ibuprofen and oramorph and he was off IV fluids. His dysuria had resolved and his eyes were no longer red and watery. Ophthalmology assessment ruled out uveitis, conjunctivitis and dry eyes. On examination, he had bilateral knee redness and swelling, bilateral ankle swelling, and an inflamed, swollen left interphalangeal joint. His hands and rest of musculoskeletal examination was unremarkable. He went on to complete a 7 day course of IV tazocin. By day 24 of illness, he was off IV antibiotics. His knee and ankle joints’ swelling had resolved, although his large left interphalangeal joint was still red, but less swollen. His parotid glands were palpable, but not visibly swollen or enlarged. He was able to eat and drink without vomiting. His stool began to thicken and were less frequent. His inflammatory markers dropped significantly (CRP 33). Over the next few days, and whilst tolerating a normal diet, off regular pain relief and anti-emetics, he completed his recovery and was discharged home. This case describes reactive arthritis with parotitis likely secondary to Shigella toxin. The diagnosis was unclear when the child developed parotitis, which is not a feature of reactive arthritis/Reiter’s syndrome. We note however, cases of reactive arthritis with parotitis reported in literature by JP Reckless in 1972 (London), Alvarez in 1989 (Spain), G Himmelreich in 1991 (Germany), and A Knecht in 1988 (Israel / no abstract available). Key learning points The main learning points in this case are that we had to go through some very old literature to elicit the connection between parotitis and reactive arthritis. Additionally, we highlight the importance of a multidisciplinary approach in managing a patient with acute illness and an initially unclear diagnosis. Input was required from general paediatrics, ENT, microbiology, rheumatology, and the paediatric surgery teams.

Introduction: Multinodular goiter (MNG) is a common thyroid condition that is usually asymptomatic. However, in rare instances where the gland enlarges substantially, it can cause significant compression of the airway and important cervical vessels. Case Presentation: A 49-year-old woman with a 10-year history of a progressively enlarging neck mass presented with symptoms of airway obstruction and bilateral great vessel compression, including dyspnea, facial swelling, and orthostatic dizziness. Imaging revealed a massively enlarged multinodular thyroid gland causing tracheal deviation and bilateral compression of the carotid arteries and jugular veins, without extension into the thoracic inlet. Thyroid function tests confirmed a euthyroid state.The patient underwent a successful total thyroidectomy. Histopathological examination showed a cystic adenomatous goiter. Postoperative recovery was uneventful except for transient hoarseness, which resolved during follow-up. Discussion: A goiter is defined as an enlargement of the thyroid gland, typically characterized by swelling in the anterior neck. It can be classified as endemic or non-endemic, diffuse or nodular, and toxic or non-toxic. This rare presentation underscores the potential severity of untreated MNG.

Vitamin C and postoperative outcomes following mandibular third molar extraction: a randomized split-mouth study.

Ghost Cell Odontogenic Carcinoma (GCOC) is a rare malignant neoplasm of odontogenic epithelium, characterized by aggressive behavior and distinctive histological features, including ghost cells and infiltrative growth. This study reports the case of an 80-year-old male patient who presented with right facial swelling and nasal obstruction. Computed tomography revealed an expansive, heterogeneous mass with cystic areas and coarse calcifications, causing bone destruction in the right maxillary region. Histopathological analysis showed odontogenic epithelial tissue, ghost cells, inflammatory infiltrate, and dentin deposits. Immunohistochemical studies confirmed an epithelioid neoplasm with atypia, mitotic figures (6/10 HPF), and diffuse positivity for cytokeratin, supporting the diagnosis of GCOC. Radiotherapy was initiated but discontinued after six sessions due to clinical deterioration, culminating in the patient’s death. This case underscores the diagnostic and therapeutic challenges of GCOC, reinforcing its aggressive nature, the role of immunohistochemistry in differentiating it from dentinogenic ghost cell tumor, and the importance of early diagnosis for improved prognosis. Keywords : Ghost cell odontogenic carcinoma, Ghost Cells, Odontogenic tumors.

Disclosure: P. Kukunoor: None. M. Katta: None. B. Korlakunta: None. S. Kotharu: None. R. Devulapally: None. H. Bunga: None. D. Korlakunta: None.Background: This case study explores a rare complication of long-standing, untreated hypothyroidism Van Wyk–Grumbach syndrome [VWGS] which is often misinterpreted due to its overlapping symptoms with other endocrine disorders. Hypothyroidism may contribute to metabolic disturbances, that may exacerbate T1DM and lead to frequent DKA episodes.[1]Case Presentation:A 12-year-old female born from a 1st degree consanguineous marriage presented with a 45-day history of progressive shortness of breath, initially grade 1, worsening to grade 4 in 2 days. The associated symptoms are chronic constipation (2-3 times/week), excessive sleepiness, and swelling of both legs. She is diagnosed with T1DM at 3 years of age with a history of frequent hospitalizations due to DKA. Menarche is attained at 10 years of age with infrequent and irregular cycles.On clinical examination, the patient had disproportionate short stature (<3rd percentile) with an upper/lower segment ratio of 1.2. Axial/pubic hair was absent with no breast bud development indicating a sexual maturation rate of A1B1P1. Other findings included a depressed nasal bridge, dry skin, pallor and non-pitting generalized edema with facial puffiness. Laboratory investigations revealed severe anemia (Hb 4.5 g%), overt hypothyroidism (markedly elevated TSH, low free T4, anti-TPO antibodies positive), hyperglycemia (serum glucose 560 mg/dL), decreased LH and increased FSH levels and hypercholesterolemia. Imaging studies revealed bilateral polycystic ovaries, significantly delayed bone age, mild pericardial effusion and fatty liver. These features of hypothyroidism, isosexual precocious puberty, and delayed bone age, collectively point toward the classic triad of VWG syndrome. The patient was given a packed red blood cell transfusion for anemia, levothyroxine therapy (37.5 mcg/day), and insulin dosage adjustments. After 2 months, the patient showed marked remission of symptoms, indicating a positive response. Continued monitoring is essential to ensure sustained recovery. Conclusion: VWGS is a rare complication of hypothyroidism, characterized by diverse manifestations and associations, posing a significant diagnostic challenge. However, being a preventable condition, this case highlights early diagnosis and prompt treatment are essential to prevent morbidity and long-term complications. Hypothyroidism can frequently occur in association with T1DM, necessitating routine thyroid screening.[1]Reference:1.Al Jabri A, Al Johar A, Yacoubi MT. A Rare association of Mauriac syndrome and Van-Wyk Grumbach syndrome found in a young Saudi girl with poorly controlled type 1 Diabetes and Hypothyroidism: A Case Report and Brief literature review. Integr J Med Sci. 2023 Jan15Presentation: Sunday, July 13, 2025

Disclosure: N. Bang: None. M. Diab: None. J. Lim: None.Introduction: Crooke cell adenoma (CCA) is an uncommon but aggressive form of corticotroph adenoma comprised of Crooke cells that secrete ACTH that contribute to Cushing disease. There have been 106 documented cases of this condition (1). Crooke cells are comprised of cytoplasmic granules containing hyaline. CCA is diagnosed when over half of the cells exhibit these cytological changes. Crooke cells are immunopositive for the transcription factors T-PIT and CK8/18, as well as for ACTH. Case Description: A 57 y/o woman with a history of primary hypothyroidism presented to the emergency department for impaired peripheral vision, fatigue, facial swelling, and weight gain. Her brain MRI revealed a pituitary macroadenoma that was 2.8 cm x 3.7 cm x 4.1 cm in size with encasement of the left internal carotid artery. The endocrinology team evaluated the patient for pituitary hormonal disorders. The morning cortisol and ACTH levels were both elevated at 19.1 mcg/dL (6.2 to 19.4 mcg/dL) and 209 mcg/dL (7.2 to 63.3 mcg/dL), respectively. The 24-hour urine cortisol level was high at 362 mcg (6 to 42 mcg/24 hr). These findings were concerning for Cushing disease. The patient subsequently underwent transsphenoidal resection of her pituitary mass. The tumor pathology revealed a corticotroph adenoma with positive staining for ACTH, CK8/18, and T-PIT. After surgery, the patient developed partial hypopituitarism leading to arginine vasopressor deficiency and secondary adrenal insufficiency. The patient is being closely monitored for either persistence versus recurrence of Cushing disease. Discussion: This case shows that it is important to consider CCA in the differential when treating patients with Cushing disease. CCA can be challenging to treat, as the recurrence rate can be greater than 60 percent (2). CCA can transform into Crooke cell carcinoma, which can contribute to dural metastases. Complications of hypercortisolism impair quality of life and increase patient’s mortality. It is crucial to understand how to identify patients who are at considerable risk of developing Crooke cell changes. However, there are currently no clinical recommendations available to guide this. Findings from this study (3) can be used to understand the management of CCA. This will make it easier for clinicians to recognize and to manage CCA in their practices. 1 Taiwo A, Kamalumpundi V, Becker N, et al. Pituitary crooke cell adenoma: Two cases of an aggressive pituitary adenoma. JCEM Case Rep. 2023;1(6): luad114. 2 Olivera CB. The 2022 WHO classification of tumors of the pituitary gland: An update on aggressive and metastatic pituitary neuroendocrine tumors. Brain Pathol. 2025;35(1): e13302. 3 Heaney AP. Clinical review: Pituitary carcinoma: Difficult diagnosis and treatment. J Clin Endocrinol Metab. 2011;96(12): 1064.Presentation: Sunday, July 13, 2025