United States

• Alpha-Gal Syndrome (AGS) is a delayed-onset IgE-mediated food allergy caused by an immune response to galactose-alpha-1,3-galactose (alpha-gal), a carbohydrate found in most mammals (e.g., pork, beef, rabbit, lamb, venison) but absent in humans. AGS is linked to bites from the lone star tick (Amblyomma americanum) in the U.S., which triggers sensitivity to alpha-gal.
• Unlike typical food allergies, AGS reactions occur 2–6 hours after consuming red meat or 4-legged mammalian derived products. Symptoms range from hives, itching, and GI distress to severe anaphylaxis.
• Alpha-gal in some medications necessitates careful prescribing to prevent allergic reactions, underscoring the need for thorough assessment and increased provider awareness.
• The CDC identified 110,000+ suspected cases (2010–2022), though the true number may be higher due to underdiagnosis and there are many healthcare providers who may not be familiar with the emerging disease1 . This study examines atrial fibrillation management in an AGS patient, highlighting allergic sensitivity to dofetilide and alternative treatment strategies.

2025 AMA Research Challenge – Member Premier Access

Challenges in Managing Atrial Fibrillation in a Patient with Alpha-Gal Syndrome: A Case of Allergic Sensitivity to Dofetilide

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Background
Dual epidural catheters (DEC) were originally described in 1949 to alleviate labor pain (1). Although uncommon, the technique has been successfully used for complex surgical procedures including spine deformity surgery, esophagectomy, and abdominal perineal resections (2,3,4). DECs have not been shown to significantly increase adverse events and have been associated with a 50% reduction in the rate of major post-operative complications, including anastomotic leaks, pulmonary complications, atrial fibrillation, and sepsis (3). We present a patient who received DEC for a two-stage removal of a coccygeal tumor.
Case Presentation
A 62-year-old 60kg female with a history of sacral chondrosarcoma s/p multiple intralesional resections presented for a 2-stage surgical removal. The first procedure was an anterior discectomy of the lumbar sacral spine. The pain service placed a T8-T9 epidural and started an infusion of bupivacaine 0.075% with hydromorphone 2mcg/cc at 5cc/hr. Patient reported 0/10 post op pain. On POD 2, she underwent stage 2 (posterior approach) of her tumor resection, and the surgeon placed a Lumbar epidural. The lumbar epidural infusion started at the post-op at 3cc/hr of the same epidural solution while the thoracic epidural was continued at 3cc/hr. Notably, the first epidural was continued intraoperatively at a rate of 3cc/hr during the second stage. After her second procedure, the patient again reported 0-3/10 pain for the next 5 days with both epidurals in place. Hemodynamics remained stable throughout, and the patient quickly met postoperative milestones. Multimodal agents added to her pain regimen included acetaminophen 500 mg q6h IV, gabapentin 300mg BID, and methocarbamol 500mg q8h IV. For breakthrough pain, her regimen included epidural boluses of 3cc q3h, oxycodone 5mg q4h prn and hydromorphone 0.25mg q4h PRN. She seldom required any PRN medications. Both catheters were removed on POD 5 from the second surgery.

Discussion
Overall, our patient tolerated the procedure and successfully transitioned to oral multimodals. The DEC technique has been reported to decrease opioid requirements and dependence on mechanical ventilation, which is increasingly important for today’s aging and sicker population. It allows for limited opioid use and its side effects such as respiratory depression, delirium, and ileus. The use of neuraxial can promote early recovery of bowel function, shortened hospital length of stay, decreased risk of perioperative complications such as immunosuppression and opioid induced endocrinopathy (4). While the DEC approach is promising, risks must be balanced with the benefit of the patients. Kinking, knotting, or looping of the catheter can cause it to break off and be left within the patient as seen in rare cases (5). However, complications of DEC are the same as single epidural catheters, and include infection, hematoma, and abscesses (6).



Dual Epidural Catheters for effective pain management after sacral coccygeal tumor removal

Abstract Title: 
Double jeopardy: A case of anomalous RCA arising from left coronary sinus with inter-arterial course complicated by significant advanced atherosclerotic disease

Background:
Coronary artery anomalies are developmental variations with an estimated incidence of 1-5.6%. While often benign, certain anomalies can lead to significant ischemic events, arrhythmias, and sudden cardiac death. The anomalous origin of a right coronary artery (RCA) from the left coronary sinus with an inter-arterial course between the aorta and pulmonary artery is a high-risk "malignant" variant. This case highlights the diagnostic and management complexities when this 
congenital anomaly is compounded by advanced acquired atherosclerotic disease

Case presentation:
A 64-year-old female with a history of hypertension, diabetes, and endstage renal disease on peritoneal dialysis presented with acute hypoxic respiratory failure, hypertensive emergency, and elevated troponins. Her hospital course was complicated by atrial fibrillation and a suspected subacute stroke. Initial electrocardiograms showed a left ventricular hypertrophy with strain 
pattern, later evolving to deep T-wave inversions. A left heart catheterization was performed for ongoing chest pain, revealing an 80% heavily calcified proximal left anterior descending artery lesion, which was treated with shockwave atherectomy and a drug-eluting stent. The RCA could not be engaged. A subsequent coronary computed tomography angiography (CTA) identified an anomalous RCA originating from the left coronary sinus. The vessel had a high-risk inter-arterial course between the aortic and pulmonary artery roots and demonstrated extensive, long-segment, 
calcified atherosclerotic plaque causing possible severe obstructive luminal narrowing. 

Discussion:
This case represents a "double jeopardy" scenario, where a high-risk congenital anomaly is superimposed with severe acquired coronary artery disease. The inter-arterial course of the RCA creates a risk for dynamic, exertional compression between the great vessels, while the fixed, calcified atherosclerotic plaque independently obstructs blood flow. This combination dramatically elevates the risk for myocardial infarction and sudden cardiac death, as even minor dynamic 
compression can become hemodynamically critical in the presence of a pre-existing stenosis. 
Determining functional significance of such lesions becomes pivotal. While surgical revascularization with coronary artery bypass grafting (CABG) is the definitive treatment to address both the dynamic and fixed obstructions when significant, patient had symptomatically improved. For further risk stratification, outpatient Stress test and Ambulatory monitoring were pursued which were negative for inducible ischemia and ventricular arrhythmias. The decision was then made to pursue aggressive medical management. This case underscores the importance of advanced imaging like coronary CTA in diagnosing complex coronary anatomy and highlights the challenges in managing patients with combined congenital and advanced acquired cardiovascular pathologies. We anticipate newer technologies like CT FFR might prove beneficial in such cases as they may provide crucial functional context to anatomical abnormalities, enabling a more comprehensive diagnostic assessment.


Double Jeopardy: An anomalous RCA with interarterial course and significant CAD

Background: Hypoglycemia is a common complication in pregnant patients with Type 1 diabetes, often exacerbated by hormonal changes and intensive insulin therapy. While mifepristone has been associated with hypoglycemia due to anti-glucocorticoid effects, the role of misoprostol, a prostaglandin E1 analog used in medical abortions, remains unclear.

Case Presentation: We describe a 28-year-old woman with well-controlled Type 1 diabetes who experienced recurrent severe hypoglycemia following misoprostol administration for medical termination of pregnancy. She had no history of hypoglycemia unawareness or adrenal insufficiency. Glycemic instability persisted for several days post-administration and required significant insulin dose reductions.

Discussion: This case highlights a possible association between misoprostol and hypoglycemia in patients with Type 1 diabetes. Proposed mechanisms include prostaglandin-mediated alterations in glucose metabolism or impairment of counterregulatory hormonal responses. A review of the literature revealed limited data on this phenomenon, underscoring the need for further investigation and to develop guidelines for managing glycemic control in similar clinical scenarios.

Conclusion: Clinicians should be aware of the potential for misoprostol-induced hypoglycemia in individuals with Type 1 diabetes. Preemptive insulin adjustments and close glucose monitoring are advised during medical management of pregnancy in this population.

Misoprostol-Induced Severe Hypoglycemia in a Type 1 Diabetic: 
A Case Report and Literature Review

Abstract Title

Use of Hypercalcemia and Vitamin D Levels in the Diagnosis of Sarcoidosis

Background
Sarcoidosis is a granulomatous disorder that most often affects the respiratory system, but can involve tissues throughout the body. The disease may cause hypercalcemia (termed sarcoidosis associated hypercalcemia (SAHC)) as macrophages within the granulomas secrete 1α-hydroxylase resulting in conversion of 25(OH)D to 1,25(OH)2D. Renal injury can be seen in sarcoidosis secondary to hypercalcemia or as a direct result of granuloma formation in the kidneys. 

Case Presentation
A 65 year old female with past medical history notable for CKD II, HTN, DMII, and asthma-COPD overlap syndrome presented to the emergency department for the second time in one month due to hypercalcemia. The patient noted mild generalized fatigue and non focal weakness. Her medications included metformin, semaglutide, and a multivitamin. Laboratory evaluation revealed Ca of 12.2, ionized Ca of 6.1, phosphorus of 7.7, PTH of 18.3, and Cr of 3.2 from a baseline of 1.0 (GFR = 56). PTHrP from her prior hospitalization earlier in month revealed 25(OH)D wnl, 1,25(OH)2D wnl, kappa/lambda ratio mildly elevated, SPEP wnl, UPEP wnl, and urine Ca wnl. Workup of humoral hypercalcemia of malignancy included CT chest, abdomen, and pelvis, and colonoscopy which were all non-revealing. On hospital day 10 a kidney biopsy was performed and eventually revealed non-necrotizing granulomas consistent with sarcoidosis. The patient was started on prednisone with slow normalization of calcium and creatine levels.

Discussion
The classic lab profile of SAHC of normal/low PTH, low 25(OH)D and high 1,25(OH)2D is actually not seen in most cases. Hypercalcemia itself is present in only 6% of patients with sarcoidosis. Elevated PTH is not consistent with SAHC and should prompt a search for a source of PTH secretion. Of patients with SAHC, one study found that 84% of patients had reduced 25(OH)D levels, with only 11% having correspondingly elevated 1,25(OH)2D levels. Potential confounders of vitamin D measurements include supplementation, dietary intake, and sun exposure. Of note, our patient previously discontinued vitamin D supplementation, but was still taking a multivitamin which contained vitamin D. Thus, in a patient with hypercalcemia and kidney injury but with an equivocal workup, low 25(OH)D can support a diagnosis of sarcoidosis, but elevated 1,25(OH)2D provides little diagnostic value. However, 25(OH)D has several confounders, and this case illustrates that it is neither completely sensitive nor specific, and biopsy is still necessary to establish a diagnosis.

Use of Hypercalcemia and Vitamin D Levels in the Diagnosis of Sarcoidosis

Introduction: Muir-Torre syndrome (MTS) is a rare phenotypic variant of Lynch syndrome with approximately 200 reported cases occurring in both sexes, having a male-to-female ratio of 3:2. It is characterized by both visceral malignancies (colorectal and endometrial tumors) and dermatological neoplasms (sebaceous adenoma, sebaceous adenocarcinoma, keratocanthomas and basal cell carcinoma with sebaceous differentiation). Sebaceous adenomas are the most common dermatological manifestations on the head and neck, particularly the periocular area. In MTS, 56% of skin lesions occur after diagnosing visceral malignancy, 6% co-occur, and 22% occur before a visceral malignancy diagnosis. MTS is most commonly associated with MSH2 mutations and rarely with MLH1 and MSH6, also found in Lynch Syndrome.
Case Description: This is the case of a 64-year-old female patient with a medical history of cecal adenocarcinoma. At the age of 60, she underwent a screening colonoscopy, which revealed a large polypoid carpeted mass. The mass was surgically removed through hemicolectomy, and the pathology reported 3.1cm intramucosal well-differentiated stage 0 adenocarcinoma. Due to a family history of endometrial carcinoma in her mother, breast cancer in her sister, and colon cancer in her cousin, subsequent genetic testing was recommended, revealing an MSH6 mutation, confirming the diagnosis of Lynch syndrome. The patient also underwent a prophylactic hysterectomy with B/L salpingo-oophorectomy. Four years later, the patient developed a lesion on her right lower eyelid, which was surgically excised and later identified as a sebaceous adenoma with MSH6 gene mutation, indicating Muir-Torre syndrome (MTS). Based on the Mayo MTS score & the MSH6 mutation, the diagnosis of MTS was supported. Later, the patient underwent a colonoscopy to rule out colon cancer, which revealed two small sessile polyps, each measuring 2 mm, in the mid-sigmoid colon.
Conclusion: This case emphasizes the importance of early identification of patients with Muir-Torre syndrome (MTS), as they are at a higher risk for developing multiple primary malignancies, including GI, gynecological, and dermatological tumors. MSH6 mutations are linked to the late onset of Lynch syndrome, which explains why the patient developed malignancy later in life. It highlights the crucial role of ophthalmologists, dermatologists, and gastroenterologists in regular cancer screening and surveillance for the timely detection and management of MTS.

Exploring Muir-Torre Syndrome: A rare case study of Eyelid Sebaceous Adenoma in a Lynch syndrome patient

Background
Anabolic androgenic steroids (AASs) are known to cause hepatotoxicity, with significant concern for inducing cholestasis and vanishing bile duct syndrome (VBDS). The risk is increased when combined with other supplements, such as the X-39 patch, containing copper. VBDS is a rarely reported but serious complication of AAS use, characterized by cholestasis and histological evidence of intrahepatic bile duct loss. Diagnostic confirmation requires a liver biopsy, showing the loss of at least 50% portal tracts. 

Case Presentation
A 27-year-old male with a history of low testosterone levels, being treated with testosterone supplementation, presented to the emergency department with symptoms of pruritus and jaundice. The patient was also reportedly using the X-39 patch for general wellness support. On physical examination, the patient appeared icteric. Laboratory studies revealed elevated liver enzymes – aspartate transaminase (AST) 152 U/L, alanine transaminase (ALT) 227 U/L, alkaline phosphatase (ALP) 216 U/L, total bilirubin level of 32.6 mg/dL, elevated WBC (17.6×109/L), and platelet count (657×109/L). His initial copper level was markedly elevated at 3,248 μg/dL. USG, MRI, and MRCP of the abdomen were unremarkable. Liver biopsy was negative for malignancy but demonstrated cholestatic hepatitis with mild focal portal chronic inflammation with stage-1 fibrosis. The infectious and autoimmune workups were unremarkable. Cytomegalovirus PCR depicted low-level viremia. The hepatitis panel was negative. Anti-nuclear antibodies were negative, with a mildly elevated anti-smooth muscle antibody. Ceruloplasmin level was within normal limits, while alpha-1 antitrypsin was slightly elevated. The patient was managed with immediate cessation of testosterone supplementation, along with penicillamine to chelate copper. Supportive care for symptom relief was provided – colestipol, hydroxyzine, and gabapentin for pruritus, and ursodeoxycholic acid for prevention of gallstone formation. Following treatment, the patient's copper level decreased to 402 μg/dL. Serial liver function tests showed gradual improvement over weeks, with normalization of bilirubin and significant reductions in AST and ALT levels. 

Discussion: 
VBDS is often a diagnosis of exclusion in patients with elevated ALP levels (≥2 times the upper limit of normal) when other causes of cholestasis are ruled out. Our case is only the fourth documented instance linking AAS use to this condition, and the first case ever to report the association of VBDS to the X-39 wellness patch, alongside AAS. While most cases improve with time, severe VBDS can lead to acute liver failure and death. Clinicians should maintain a high index of suspicion for drug-induced liver injury, particularly in patients using testosterone and wellness supplements.

Testosterone-induced Vanishing bile duct syndrome associated with X-39 wellness patch: A rare case report

This is a case of a patient who underwent an elective laparoscopic cholecystectomy after previously presenting with acute cholecystitis. His operation was complicated by a high common hepatic duct (CHD) injury requiring complex surgical repair. This case highlights the importance of early recognition of this type of injury and the utility of a Kasai portoenterostomy as an operative repair in high CHD injuries in the absence of a viable extrahepatic duct viable for repair.

Surgical Management of a Complex High Hepatic Duct Injury: A Case Report

Diaphragmatic hernias result from a defect in the diaphragm that allows abdominal contents to herniate into the thoracic cavity. The most common cause is acute trauma, particularly blunt force trauma, more than penetrating trauma. Congenital causes are the second most common cause. Such as Bochdalek and Morgagni hernias. There are other types and the most uncommon is a spontaneous diaphragmatic hernias (SDH). This paper presents a novel case of SDH, leading to gastric fundus protrusion and incarceration, resulting in abscess formation. The patient’s medical history did not suggest trauma or congenital anomalies, making this case an acquired SDH, with fewer than 40 documented occurrences. Diagnosis is often delayed due to the overlap of symptoms with other conditions, and SDH frequently presents with nonspecific gastrointestinal or respiratory symptoms. Imaging, particularly CT, is crucial for diagnosis. The patient underwent successful repair using a transabdominal robotic approach with the Da Vinci Xi system, marking the first documented case of SDH repaired exclusively with robotic techniques. This case highlights the importance of considering SDH in patients with unexplained thoracoabdominal pain, particularly in the absence of trauma, and demonstrates the efficacy of robotic surgery in minimizing complications and reducing morbidity in such cases.

Trans-abdominal Approach in a Spontaneous Diaphragmatic Hernia: A Case Report

Introduction
Intracholecystic papillary neoplasm (ICPN) is a rare, visibly distinct lesion arising
from the gallbladder lining and projecting into its lumen. It is often discovered
incidentally when asymptomatic but may sometimes cause symptoms such as pain,
jaundice, weight loss, or ascites. ICPN is very rarely reported in Nepal, highlighting
the need for careful evaluation and management.
Presentation of case
We report a 63-year-old man diagnosed incidentally with ICPN during routine
evaluation. The patient had no symptoms such as abdominal pain. He underwent
laparoscopic cholecystectomy and recovered well without complications.
Discussion
ICPN is a preinvasive gallbladder tumor that can show different histologic patterns,
typically appearing as papillary or polypoid growths. Imaging like ultrasound and CT
can suggest ICPN, but a definitive diagnosis requires surgical removal and
microscopic confirmation. Intraoperative frozen section assessment of tumor invasion
and margins is helpful for surgical decision-making, as demonstrated in this case.
Early diagnosis and surgery generally result in better outcomes than those seen in
invasive gallbladder cancer.
Conclusion
Because ICPN is rare and often presents subtly, a thorough clinical workup with
appropriate imaging is essential. Early surgical intervention supported by
intraoperative pathology evaluation can improve prognosis and patient outcomes.

An incidental finding of Intracholecystic Papillary Neoplasm- A rare case report

Background: 
Extramedullary plasmacytoma (EMP) is a rare form of plasma cell neoplasm characterized by monoclonal plasma cell infiltration without systemic evidence of multiple myeloma. It represents 1.6-4% of all plasma cell tumors. The most common sites of involvement are the upper respiratory and gastrointestinal tracts, while anterior chest wall involvement is rare and can mimic other common thoracic malignancies. Here, we report a case of a 50-year-old man presenting with a large anterior chest wall mass diagnosed as extramedullary soft tissue plasmacytoma occurring years after blunt thoracic trauma. Plasmacytoma should be considered in the differential diagnosis of anterior chest wall masses, especially when associated with a history of trauma and presence of invasive features on imaging. A combination of clinical presentation, imaging, and biopsy is critical for diagnosing atypical soft tissue neoplasms.
Case Presentation: 
A 50-year-old man presented with a progressively enlarging anterior chest wall mass, initially noticed 5–6 years after blunt thoracic trauma. The mass remained stable for years but rapidly increased in size over six months, causing dyspnea, dysphagia, and odynophagia. Physical examination revealed an 18 cm, firm, non-mobile mass with superficial venous prominence. The initial differentials included thymoma, teratoma, thyroid neoplasm, and lymphoma. However, the history of trauma, chronic presentation, recent progression, and normal tumor markers raised suspicion for a soft tissue or bony lesion. Contrast-enhanced CT showed a lobulated, calcified mass in the superoanterior mediastinum with scattered enhancement, sternal erosion, invasion of the first rib, anterior chest wall extension, and tracheal compression. Laboratory workup revealed hemoglobin 11.6 g/dL, calcium 9 mg/dL, creatinine 0.8 mg/dL, and normal TSH, LDH, β-hCG, and AFP levels, but urine electrophoresis demonstrated an M spike with significantly elevated free kappa light chains (1956.2 mg/L). Histopathology showed diffuse sheets of plasma cells with eosinophilic cytoplasm and eccentric nuclei. Immunohistochemistry was positive for CD138, MUM1, CD56, and kappa light chain, consistent with a diagnosis of kappa-restricted extramedullary plasmacytoma.
Discussion: 
This case demonstrates a rare post-traumatic extramedullary plasmacytoma with extensive local invasion, highlighting the need to consider plasma cell neoplasms in the differentials of anterior mediastinal masses. In this case, scar tissue from prior injury may have created a permissive microenvironment, allowing delayed plasma cell proliferation. Imaging revealed aggressive features suggestive of soft tissue plasmacytoma, and biopsy confirmed the presence of plasma cells. While radiotherapy is the primary treatment modality, chemotherapy may also be used in combination based on the tumor characteristics.


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Dual Epidural Catheters for effective pain management after sacral coccygeal tumor removal

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