Whipple's Disease Endocarditis Following Immunomodulatory Treatment for Arthritis: A Case Report and Screening Recommendation.

Whipple's disease (WD) is a rare systemic disorder affecting various organ systems, including the gastrointestinal, cardiovascular, and joint systems. This report discusses a case of WD endocarditis likely associated with tocilizumab (TCZ), an immunomodulator used to treat refractory seronegative arthritis, in a patient with coronary artery disease and rheumatoid arthritis. The diagnosis was confirmed through laboratory studies, imaging, and esophagogastroduodenoscopy with biopsies. WD is increasingly recognized as a potential etiology of seronegative arthritis, with joint pain often preceding gastrointestinal symptoms. Immunomodulatory agents such as TCZ, while effective for rheumatoid arthritis, may exacerbate underlying WD, potentially leading to severe complications such as endocarditis. This case reveals the importance of considering WD in patients with refractory arthritis and the necessity of thorough diagnostic evaluation before initiating immunomodulatory therapy. Epidemiological studies indicate a higher prevalence of WD in certain demographics, highlighting the need for targeted screening with noninvasive screening methodologies, such as stool and saliva polymerase chain reaction testing.

A rare case of Whipple disease presenting as a hydrosalpinx and granulomatous peritonitis.

INTRODUCTION: Whipple disease is a rare infectious disease caused by the bacterium Tropheryma whipplei. The classic form affects gastrointestinal and musculoskeletal systems; but other forms may damage the heart, brain, or lungs. Due to non-specific and diverse clinical symptoms, diagnosis of Whipple disease is challenging and often late. Adequate and timely antibiotic treatment is essential for favorable outcome. CASE PRESENTATION: Here we present a case of a young woman admitted to the gynecological clinic for diagnostic laparoscopy for suspected haemato-/hydro- salpinx and peritoneal endometriosis. Macroscopic findings during laparoscopy revealed miliary whitish lesions in the pelvis and histopathology reported granulomatous salpingitis and peritonitis. She was complaining of intermittent abdominal pain, bloating and weight loss. Subsequently, the laparoscopy symptoms worsened and her general condition deteriorated. Differential diagnosis included infective agents such as Mycobacterium tuberculosis; in addition to sarcoidosis, granulomatosis with polyangiitis, and malignancies; all of which were excluded. Finally, Tropheryma whipplei was suspected, and after esophagogastroduodenoscopy with duodenal biopsy, long-term antibiotic treatment was initiated and the patient fully recovered. CONCLUSIONS: Although Whipple disease is rare, it is important to have a high level of awareness for Tropheryma whipplei infection. The localization and course of Whipple's disease may be unpredictable, but a favorable outcome is expected with adequate antibiotic treatment.

Pylorus-preserving pancreaticoduodenectomy with superior mesenteric vein resection and reconstruction in a child with recurrent hepatoblastoma after liver transplantation.

Pancreaticoduodenectomy with vascular reconstruction is rarely performed in children. We present a 3-year-old male with stage IV hepatoblastoma and pre-treatment extent of disease (PRETEXT) stage III with tumor into the portal vein and superior mesenteric vein (SMV), and with brain and lung metastases status post chemotherapy and stereotactic radiosurgery to left frontal brain lesion. He then underwent deceased donor liver transplant with Roux-en-Y hepaticojejunostomy complicated by two recurrences to bilateral lungs treated with wedge resections. His course lastly involved a third hepatoblastoma recurrence to the SMV that was managed with pylorus-preserving pancreaticoduodenectomy with SMV resection and reconstruction.

Needle Knife Stricturoplasty for Hepaticojejunostomy Stricture After Whipple Procedure.

Hepaticojejunostomy anastomotic stricture is a relatively uncommon postoperative complication after a Whipple procedure. However, they are increasingly being observed because of advancements in operative mortality rates and the widening of operative indications to include benign diseases such as chronic pancreatitis and intraductal papillary mucinous neoplasm. In this article, we describe a patient diagnosed with intraductal papillary mucinous neoplasm and another with pancreatic cancer, both of whom developed jaundice after undergoing the Whipple procedure. Subsequent investigation revealed a benign hepaticojejunostomy anastomotic stricture, successfully treated with stricturoplasty using a needle knife.

Duodenal obstruction in a 38-year-old: Case report of an unusual ovarian cancer presentation requiring a Whipple procedure.

Since ovarian cancer typically spreads intraperitoneally or via lymphatics, a retroperitoneal duodenal obstruction is a rare presentation of ovarian cancer. Such upper gastrointestinal obstruction in a young patient is diagnostically challenging and surgically difficult to address. In this case report, we describe that in an interdisciplinary approach a Whipple pancreaticoduodenectomy could be safely implemented into the interval debulking surgery to achieve complete cytoreduction. No postoperative complications were encountered. The surgical procedure was able to remove the upper gastrointestinal obstruction and thereby the need for a venting gastrostomy tube and total parenteral nutrition and thus provided good quality of life and additional lifetime.

Varices at Choledochojejunostomy Site as the Elusive Gastrointestinal Bleeding Source in a Post-Whipple Patient.

Ectopic varices are portosystemic collaterals that form outside the esophagogastric region. If left untreated, they can carry a high mortality rate. Although ectopic variceal bleeding occurs rarely, it continues to pose both diagnostic and therapeutic challenges and remains an under-recognized source of gastrointestinal (GI) hemorrhage. We present a case of GI bleeding from varices formed at the choledochojejunosomy site in a post-Whipple patient. This case describes the rare finding of ectopic varices after pancreaticoduodenectomy, emphasizes the challenges in diagnosis and treatment, and highlights the need for a multidisciplinary approach managing GI bleeding in this patient population.

Outcomes of minimally invasive vs. open pancreatoduodenectomies in pancreatic adenocarcinoma: analysis of ACS-NSQIP data.

INTRODUCTION: Pancreatoduodenectomy (PD) for pancreatic adenocarcinoma (PDAC) presents a significant challenge owing to its aggressive nature. Traditionally performed as open surgery, the advent of minimally invasive surgery (MIS) including laparoscopic and robotic techniques, offers a potential alternative. This study assessed the use and outcomes of MIS and open PD for PDAC treatment. METHODS: We analyzed ACS-NSQIP data (2015-2021) using regression models to compare patient outcomes across open PD, MIS PD, and conversions from MIS to open (MIS-O). RESULTS: Of 19,812 PDAC patients, 1,293 (6.53%) underwent MIS, 18,116 (91.44%) underwent open PD, and 403 (2.03%) underwent MIS converted to open PD (MIS-O). The MIS rate increased from 6.1% to 9.2%. Black patients had a higher MIS-O rate (RR, 1.55; p = 0.025). Open PD was associated with more severe conditions (ASA ≥ III, malnutrition) and prior radiation therapy. MIS patients more often had neoadjuvant chemotherapy. Complex procedures, such as vein resection, favored open PD. Need for arterial resection was associated with MIS-O (RR, 2.11; p = 0.012), and operative time was significantly associated with MIS (OR: 4.32, 95% CI: 3.43-5.43, p-value: < 0.001) No differences in the overall morbidity or 30-day mortality were observed. MIS led to shorter stays but higher risks of reoperation and pulmonary embolism. MIS-O increased the delayed gastric emptying rate (RR, 1.79; p < 0.001). CONCLUSION: During 2015-2021, an increasing number of patients with PDAC are undergoing MIS PD. Morbidity and mortality did not differ between open and MIS PD. MIS was performed more frequently in patients with better nutritional status and lower ASA, or when vascular resection was not anticipated. In well selected patients, short-term outcomes of MIS and open PD seem similar.

Perioperative Spinal Cord Injury in the Setting of Whipple Procedure for Pancreatic Adenocarcinoma in a Patient With a History of Severe Cervical Stenosis.

Perioperative spinal cord injury (POSCI) is a form of traumatic acute spinal cord injury (TSCI) in the perioperative setting that is a rare but feared complication associated with severe morbidity and mortality, often resulting in significant functional impairment and significant healthcare costs for the patient. Here, we present a case report of a 65-year-old male with a past medical history of hypertension (HTN), type-2 diabetes mellitus (T2DM), stage 4 chronic kidney disease (CKD4) with a one-year history of anorexia, weight loss, jaundice, and right lower quadrant (RLQ) pain. He underwent an endoscopic ultrasound, which showed pancreatic atrophy, marked dilation of the main pancreatic duct, and a poorly defined pancreatic head mass. The patient underwent a successful pancreaticoduodenectomy and was extubated in the operating room and transferred to the surgical intensive care unit (SICU) on an oxygen face mask without complication. Four hours later it was noted that the patient's neurological exam had acutely changed with loss of motor and sensory function from the C7 dermatome down. The patient remained stable from a cardiopulmonary standpoint, and he was urgently transferred for emergency imaging of his brain and spinal cord, which demonstrated evidence of chronic spinal canal stenosis, complete cord flattening at the C5 level with profound cord edema centered at the C5 level extending from C3 to T1. Following diagnosis, neurosurgery was consulted at the SICU and a comprehensive neurological exam was performed. It was determined the patient had a grade A injury via the American Spinal Injury Association (ASIA) Impairment Scale and required an emergency cervical laminectomy. The patient was taken back to the operating room (OR) and an open cervical laminectomy was performed from C3 to C7 without any intraoperative complications. The patient was managed by a multidisciplinary SICU team for both his pancreaticoduodenectomy, perioperative traumatic acute spinal cord injury, and subsequent multilevel cervical laminectomies. The patient had a purposeful neurological recovery over the following weeks and was ultimately discharged to an inpatient physical rehabilitation facility.

A Complex Case of Obstructive Jaundice in a Septuagenarian: Diagnostic Challenges and Therapeutic Strategies.

Obstructive jaundice occurs when an obstruction in the bile duct system prevents bile from flowing from the liver into the intestine, accumulating bilirubin in the blood. This condition can result from various causes, including gallstones, tumors, or inflammation of the bile ducts. The management of obstructive jaundice depends on the underlying cause (malignant obstructions such as cholangiocarcinoma or pancreatic cancer), indicating the need for surgical intervention. The Whipple procedure (pancreaticoduodenectomy) is the standard curative approach for resectable distal common bile duct (CBD) adenocarcinoma. Doctors usually recommend adjuvant chemotherapy to reduce the risk of recurrence. We report the case of a 70-year-old male with a history of untreated hypertension, type 2 diabetes, and long-term smoking, who presented with classic signs of obstructive jaundice, including yellowing of the eyes, itching, right upper quadrant pain, and intermittent fevers. Laboratory findings revealed elevated inflammatory markers, bilirubin, liver enzymes, and leukocyte count, indicative of an inflammatory and obstructive biliary condition. Imaging studies confirmed a distal CBD stricture, including abdominal ultrasound, computed tomography scans, and endoscopic retrograde cholangiopancreatography (ERCP). Brush cytology obtained during ERCP revealed a well-differentiated adenocarcinoma of the distal CBD. The patient's treatment plan included preoperative optimization, surgical resection via the Whipple procedure, and postoperative adjuvant therapy. This case emphasizes the importance of a thorough diagnostic workup and a multidisciplinary treatment strategy in managing complex cases of obstructive jaundice in the elderly, highlighting the need for personalized care to achieve optimal outcomes.

[Bilateral panuveitis in Whipple's disease: Case report]. / Panuvéite bilatérale dans la maladie de Whipple : à propos d'un cas.

Whipple's disease is a rare disease linked to chronic infection with the intracellular gram-positive bacterium, Tropheryma whipplei. The clinical signs suggestive of this disease are the association of unexplained fever, lymphadenopathy, gastroenterological disorders (malabsorption) and inflammatory joint disorders (arthritis). However, isolated cardiological, neurological or ophthalmological forms have been described. We report the rare case of a 56-year-old patient complaining of floaters and recent visual loss, who presented with bilateral panuveitis in the absence of any systemic disorder. Clinical examination showed inflammation of the anterior segment, vitritis, inflammatory optic disc edema, focal retinitis, and venous vasculitis in both eyes. We describe the clinical characteristics and ancillary findings of the disease (fundus photos, visual field, auto-fluorescence, macular OCT, fluorescein and indocyanine green angiography). The diagnosis was made with the blood (T. whipplei) PCR test and with the help of accessory salivary gland biopsies. We describe the work-up leading to the diagnosis of Whipple's disease, the laboratory tests, and the recommended extended work-up. The patient's course was marked by complete resolution of the symptoms and clinical signs within a few months following corticosteroid therapy (1mg/kg/day) combined with hydroxychloroquine (600mg per day for 1 year) and life-long doxycycline therapy (200mg per day). In conclusion, this is a rare disease which should be discussed when dealing with steroid-resistant and/or steroid-dependent chronic uveitis with a negative work-up (especially in the presence of joint and/or digestive involvement).

Open vs robotic-assisted pancreaticoduodenectomy, cost-effectiveness and long-term oncologic outcomes: a systematic review and meta-analysis.

BACKGROUND: Whipple pancreaticoduodenectomy (PD) is a complex gastrointestinal surgery that is performed increasingly via minimally invasive approach through robotic platforms. We sought to provide a comparative review of available data regarding robot-assisted vs open PD in terms of cost-effectiveness, overall survival, and other perioperative and long-term oncologic outcomes. METHODS: Using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses criteria, PubMed, Scopus, and Web of Science databases were searched from 1980 to April 2024 using designated keywords. English-language studies comparing costs and oncologic outcomes of robotic vs open PDs were considered for inclusion. Reviews, abstracts, case reports, letters to the editor, and non-English articles were excluded. RESULTS: A total of 1733 studies were initially identified throughout the literature search. After the removal of duplicates, title and abstract screening identified 16 studies that were included in the review. No statistically significant differences were detected in terms of short-term complications (95% CI, 0.805-1.096; P = .42), mortality (95% CI, 0.599-1.123; P = .21), and readmission (95% CI, 0.959-1.211; P = .20) among patients undergoing open vs robotic PD. Robotic PDs was associated with a slightly better overall survival (95% CI, 1.020-1.233) and higher costs (95% CI, 0.134-1.139; P = .013). Mean length of stay (LOS) was higher in the open PD group (95% CI, -0.353 to 0.189; P < .001). CONCLUSION: Robotic-assisted PD had a slightly shorter LOS and improved overall survival. There were no differences in short-term complications, mortality, or readmission. The use of cohort studies and residual potential selection bias necessitate randomized controlled trials to define the benefit of robotic PD.

Prevalence of autoimmune pancreatitis in pancreatic resection for suspected malignancy: a systematic review and meta-analysis.

BACKGROUND/OBJECTIVES: Autoimmune pancreatitis (AIP) is a diagnosis-challenging disease that often mimics pancreatic malignancy. Pancreatic resection is considered to be a curative treatment for pancreatic ductal adenocarcinoma (PDAC). This meta-analysis aims to study the incidence of AIP in patients who have undergone pancreatic resection for clinical manifestation of cancer. METHODS: A comprehensive search was conducted in three databases, PubMed, Embase and the Cochrane Library, using the terms 'autoimmune pancreatitis' and 'pancreatic resection' and supplemented by manual checks of reference lists in all retrieved articles. RESULTS: Ten articles were included in the final analysis. 8917 pancreatic resections were performed because of a clinical suspicion of pancreatic cancer. AIP accounted for 140 cases (1.6%). Type 1 AIP comprised the majority of cases, representing 94% (132 cases), while type 2 AIP made up the remaining 6% (eight cases) after further classification. AIP accounted for almost 26% of all cases of benign diseases involving unnecessary surgery and was overrepresented in males in 70% of cases compared to 30% in females. The mean age for AIP patients was 59 years. Serum CA 19 - 9 levels were elevated in 23 out of 47 (49%) AIP patients, where higher levels were detected more frequently in patients with type 1 AIP (51%, 22 out of 43) than in those with type 2 AIP (25%, 1 out of 4). The sensitivity of IgG4 levels in type 1 AIP was low (43%, 21/49 patients). CONCLUSION: Even with modern diagnostic methods, distinguishing between AIP and PDAC can still be challenging, thus potentially resulting in unnecessary surgical procedures in some cases. Serum CA 19 - 9 levels are not useful in distinguishing between AIP and PDAC. Work must thus be done to improve diagnostic methods and avoid unnecessary complicated surgery.

Initial Experience of Pancreaticoduodenectomy in a Newly Developed Hepato-Pancreato-Biliary Unit Serving in a Lower-Middle-Income Country.

Introduction: Pancreaticoduodenectomy (PD) is the only potentially curative treatment for pancreatic head adenocarcinoma. This study aimed to determine the short-term outcomes of PD performed over 1 year at a newly established hepato-pancreatico-biliary unit in Khyber Pakhtunkhwa province of Pakistan. Material and Methods: A retrospective analysis of a prospectively maintained hospital information system (HIS) was undertaken of all patients referred to the unit between May 2021 and August 2022. Data were collected from the medical records of patients in the HIS. Data were analyzed for primary location, age, complications, and operative parameters. Results: The primary sites of disease were ampulla (n = 18, 52.9%), pancreas (n = 11, 32.4%), and duodenum (n = 5, 14.7%). The median duration of surgery was 7 h. 16 (47.1%) patients required blood transfusion either intraoperatively or in the perioperative period. Patients with pre-operative biliary drainage (PBD) were more likely to have multidrug-resistant positive bile cultures with a P-value of 0.2 (n = 12 [35.3%] vs. n = 5 [14.7%]). Overall morbidity was 38.2%. The most common complications were wound infection (n = 12, 35.3%), delayed gastric emptying (n = 6, 17.6%), and type B pancreatic fistula (n = 3, 8.8%). The complication rate was higher in patients with biliary stenting (n = 11 [32.4%] vs. n = 2 [5.9%]; P = 0.06). The median length of hospital stay for patients without complications was less (6 vs. 12 days; P < 0.001). The complication rate was lower in total laparoscopic PD (TLPD) with P = 0.4 (TLPD: 2.9%, open: 23.5%, laparoscopic assisted: 11.8%). 90-day mortality was zero. Conclusion: Short-term outcomes for PD in our facility are comparable to high-volume centers. PBD can significantly increase operative time, hospital stay, and morbidity.

Market Factors, Not Quality, Influence Reimbursement for Pancreaticoduodenectomy in an Era of Price Transparency.

BACKGROUND: The Centers for Medicare and Medicaid Services (CMS) price transparency rule tries to facilitate cost-conscious decision-making. For surgical services, such as pancreaticoduodenectomy (PD), factors mediating transparency and real-world reimbursement are not well described. METHODS: The Leapfrog Survey was used to identify United States hospitals performing PD. Financial and operational data were obtained from Turquoise Health and CMS Cost Reports. Chi-square tests and modified Poisson regression evaluated associations with reimbursement disclosure. Two-part logistic and gamma regression models estimated effects of hospital factors on commercial, Medicare, and self-pay reimbursements for PD. RESULTS: Of 452 Leapfrog hospitals, 295 (65%) disclosed PD hospital or procedure reimbursements. Disclosing hospitals were larger (beds > 200: 81.0% vs. 71.3%, p = 0.04), reported higher net margins (0.7% vs. - 2.1%, p = 0.04), more likely for-profit (26.1% vs. 6.4%, p < 0.001), and teaching-affiliated (82.0% vs. 65.6%, p < 0.001). Nonprofit status conferred hospitalization reimbursement increases of $8683-$12,329, while moderate market concentration predicted savings up to $5066. Teaching affiliation conferred reimbursement increases of $4589-$16,393 for hospitalizations and $644 for procedures. Top Leapfrog volume ratings predicted an increase of up to $7795 for only Medicare hospitalization reimbursement. CONCLUSIONS: Nondisclosure of hospital and procedural reimbursements for PD remains a major issue. Transparency was noted in hospitals with higher margins, size, and academic affiliation. Factors associated with higher reimbursement were non-profit status, academic affiliation, and more equitable market share. Reimbursement inconsistently tracked with PD quality or volume measures. Policy changes may be required to incentivize reimbursement disclosure and translate transparency into increased value for patients.

Pancreaticoduodenectomy in Patients With Coeliac or Superior Mesenteric Artery Stenosis: A Review of the Literature.

Pancreaticoduodenectomy (Whipple's procedure) is a technically demanding operation performed for malignant and premalignant conditions of the pancreatic head, duodenum and bile duct. Awareness of the vascular anatomy, variations, and pathology of this area is essential to achieve safe surgery and good outcomes. The operation involves division of the gastroduodenal artery (GDA) which provides communication between the foregut and midgut blood supply. In patients with coeliac or superior mesenteric artery (SMA) stenosis, this can lead to reduced blood supply to the foregut or midgut organs, with consequent severe ischaemic complications leading to significant morbidity and mortality. Coeliac artery stenosis is caused by median arcuate ligament syndrome (MALS) in the majority of patients with atherosclerosis being the second most common cause. SMA stenosis is much less common and is caused in the majority of cases by atherosclerosis. A review of preoperative imaging and intraoperative gastroduodenal artery clamp test is important to identify cases that may need additional procedures to preserve the blood supply. In this paper, we present a literature review for studies reporting patients undergoing Whipple's operation with concomitant coeliac axis stenosis (CAS) or SMA stenosis. Analysis of causes of stenosis or occlusion, prevalence, risk factors, different management strategies and outcomes was conducted.

Tropheryma whipplei Colonization in Adults and Children: A Prospective Study.

We conducted a prospective cohort study at the IRCCS Sacro Cuore Don Calabria Hospital in Negrar di Valpolicella from 2019 to 2021 to investigate the duration of T. whipplei colonization. In addition, the correlation between persistent colonization and the continent of origin, current treatment regimen, clinical manifestations, and parasite coinfection was evaluated. The cohort included subjects who were tested in a previous study (years 2014-2016) and found to be positive for T. whipplei DNA in fecal samples. Thirty-three subjects were enrolled in a prospective study between 2019 and 2021. Feces, saliva, urine, and blood were collected at baseline and after 12 months. Medical history, current treatment, and symptoms were recorded. Among them, 25% showed persistent intestinal or oral colonization, 50% had no colonization at both visits, and 25% had intermittent colonization. No association was found between persistent T. whipplei colonization and subjects' continent of origin, current treatment regimen, initial clinical manifestations, and parasite coinfection. The longest duration of persistent T. whipplei intestinal colonization exceeded six years, with 11 subjects presenting persistent positivity for more than three years, including 1 minor. Our research was limited by the lack of a strain-specific identification of T. whipplei that made it impossible to distinguish between persistence of the same T. whipplei strain, reinfection from household exposure, or infection by a new strain. Larger prospective studies are needed to further explore the implications of this persistence and determine the key factors influencing the duration of colonization and its potential health impacts.

Primary pancreatic hydatid cyst: A rare case report and diagnostic challenges.

Key Clinical Message: In cystic lesions of the pancreas, hydatid cyst should be considered in the differential diagnoses and its presence should be ruled out before any invasive interventions. Serological tests along with imaging studies related to hydatid cyst diagnostic indicators should be performed in people who live in Echinococcus granulosus endemic areas and suffer from cystic lesions of the gastrointestinal tract. Abstract: Primary pancreatic hydatid cysts, caused by the tapeworm Echinococcus granulosus, represent a rare occurrence often challenging to diagnose due to their similarity to other pancreatic conditions. This case report outlines a 67-year-old male presenting with jaundice and cholestasis but lacking typical symptoms associated with pancreatic hydatid cysts. Laboratory findings revealed elevated bilirubin levels, liver enzyme abnormalities, and tumor markers, prompting imaging studies that indicated a cystic mass near the pancreatic head. Misdiagnosed initially as a mucinous cystic neoplasm, the patient underwent Whipple surgery, unveiling a large cystic lesion upon examination.

Benign Etiology for High-Risk Intraductal Papillary Mucinous Neoplasm: A Case Report and Literature Review.

Intraductal papillary mucinous neoplasms are relatively common and entail a variable risk of malignant potential. The Fukuoka guidelines present criteria for the risk of malignant transformation and are used for risk stratification and treatment decision-making. However, these guidelines entail some fallibility with limited sensitivity and specificity. In this case, we present an individual who had many of the hallmarks of malignant transformation but was found to have no evidence of malignancy or high-grade dysplasia. We discuss the suspected etiology of this individual's condition and how it might arise in others, as well as a brief review of the literature on risk factors in intraductal papillary mucinous neoplasms.

Two cases demonstrate an association between Tropheryma whipplei and pulmonary marginal zone lymphoma.

BACKGROUND: Marginal zone lymphomas of mucosa-associated lymphatic tissues (MZL of MALT) are a group of indolent B-cell neoplasms, which are thought to arise from chronic antigenic stimulation of B-cells either due to underlying chronic infection or autoimmune disease. Little is known about potential causative pathogens in pulmonary MZL (PMZL), although some data suggests a potential role of Achromobacter (A.) xylosoxidans. METHODS: An index case of chronic pulmonary colonisation with Tropheryma (T.) whipplei and subsequent development of PMZL was identified by T. whipplei specific PCR and metagenomic next genome sequencing (mNGS). This case prompted a retrospectively conducted analysis of T. whipplei-specific PCRs in lung tissue from PMZL patients (n = 22), other pulmonary lymphomas, and normal controls. Positive results were confirmed by mNGS. A systematic search for T. whipplei and A. xylosoxidans in our in-house mNGS dataset comprising autopsy lungs, lung biopsies and lung resection specimens (n = 181) was subsequently performed. RESULTS: A 69-year-old patient presented with weight loss and persistent pulmonary consolidation. Subsequent mNGS analysis detected T. whipplei in the resected lung specimen. An antibiotic regimen eventually eliminated the bacterium. However, the consolidation persisted, and the diagnosis of PMZL was made in a second lung resection specimen. A second case of T. whipplei-associated PMZL was subsequently detected in the retrospectively analysed PMZL cohort. Both cases showed comparatively few mutations and no mutations in genes encoding for NF-κB pathway components, suggesting that T. whipplei infection may substitute for mutations in these PMZL. None of the samples in our in-house dataset tested positive for T. whipplei. In contrast, A. xylosoxidans was frequently found in both autopsy lungs and lung biopsy / resection specimens that were not affected by PMZL (> 50%). CONCLUSIONS: Our data suggests that T. whipplei colonisation of lungs may trigger PMZL as a potential driver. Systematic analyses with larger cohorts should be conducted to further support this hypothesis. The frequent detection of A. xylosoxidans in lung tissue suggests that it is a common component of the pulmonary microbiome and therefore less likely to trigger lymphomas.

Sweet Saboteur: Insulinoma Presenting As Recurrent Hypoglycemic Seizures.

Insulinoma, a rare neuroendocrine tumor of the pancreas, often presents diagnostic challenges due to its diverse clinical manifestations. We present the case of a 25-year-old female with recurrent hypoglycemic seizures and neuroglycopenic symptoms, ultimately diagnosed with insulinoma. Despite an initial asymptomatic period, the patient experienced progressively worsening symptoms over three years, culminating in eight episodes of generalized tonic-clonic seizures per week. Biochemical investigations during hypoglycemic episodes revealed elevated C-peptide and insulin levels, consistent with endogenous hyperinsulinemia. Imaging studies, including contrast-enhanced computed tomography (CECT) and Ga-DOTATATE scan, confirmed a hyper-enhancing lesion in the distal body of the pancreas, indicative of insulinoma. Histopathological examination (HPE) further corroborated the diagnosis. Prompt recognition and surgical excision led to the complete resolution of symptoms and improved long-term prognosis. This case underscores the importance of considering insulinoma in young individuals presenting with recurrent hypoglycemic episodes and highlights the significance of early diagnosis and intervention in preventing morbidity and mortality associated with this condition.