Diffuse Ulceration Research Articles

Association Between SARS-CoV-2 Infection and Acute Ischemic Colitis.

Ischemic colitis appears to be a rare but serious complication of COVID-19. About 33.3% hospitalized COVID-19 patients who underwent endoscopy showed features resembling ischemic colitis. The aim of this prospective study was to describe the symptoms, treatment, and outcomes of these patients, particularly their colonoscopy and histologic findings. We conducted a prospective study on ischemic bowel disease associated with COVID-19 across four centers from December 2022 to January 2023. All cases were identified through a comprehensive search of electronic medical records for procedure-related data. The initial diagnosis of ischemic bowel disease was confirmed using colonoscopy and biopsy findings. After the patients were discharged, a 12-month follow-up was conducted through regular phone interviews to assess their clinical outcomes. Overall, the study included 3 male patients and 9 female patients (age range, 33-76 years). Abdominal pain and hematochezia always occurred within 2weeks after COVID-19 infection (average 5.5 days). Gastrointestinal manifestations did not parallel the severity of COVID-19 infection. The descending colon was the most susceptible segment, which was involved in 10 patients (83.33%). Colonoscopy revealed diffuse redness, edema, bleeding, erosion, and ulceration of the intestinal mucosa, similar to the findings of ischemic colitis, and biopsy revealed crypt atrophy, reduction, and interstitial bleeding. All patients were self-limited without converting to chronic changes in the next 12 months. SARS-CoV-2 infection may induce transient acute colon ischemia within 2weeks, accompanied by severe clinical symptoms such as acute abdominal pain and hematochezia, which are self-limiting and do not lead to chronic symptoms.

Severe ulcerative colitis induced by COVID-19 vaccination

A 37-year-old woman developed severe colitis with diffuse mucosal erythema and ulcerations throughout the entire colon after the 3rd vaccination of COVID-19. Stool culture was negative, and the pathological findings showed increased lymphoplasmacytic and neutrophilic infiltration in the colonic lamina propria, which were consistent with ulcerative colitis. After the treatment with anti-tumor necrosis factor-α agent, the ulceration markedly improved with development of severe colonic stenosis, which was successfully dilated with endoscopic balloon dilation. In case of COVID-19 vaccination, it should be noted that vaccination could be a trigger for the onset of UC.

Epigenetic regulation and therapeutic strategies in ulcerative colitis.

Ulcerative colitis (UC) is an inflammatory bowel disease, and is characterized by the diffuse inflammation and ulceration in the colon and rectum mucosa, even extending to the caecum. Epigenetic modifications, including DNA methylations, histone modifications and non-coding RNAs, are implicated in the differentiation, maturation, and functional modulation of multiple immune and non-immune cell types, and are influenced and altered in various chronic inflammatory diseases, including UC. Here we review the relevant studies revealing the differential epigenetic features in UC, and summarize the current knowledge about the immunopathogenesis of UC through epigenetic regulation and inflammatory signaling networks, regarding DNA methylation, histone modification, miRNAs and lncRNAs. We also discuss the epigenetic-associated therapeutic strategies for the alleviation and treatment of UC, which will provide insights to intervene in the immunopathological process of UC in view of epigenetic regulation.

Social Media Trends, Dry Scooping, and Extensive Esophageal Ulcerations.

We describe a case of severe odynophagia and dysphagia caused by dry scooping of multi-ingredient pre-workout powder (MIPS) with diffuse esophageal ulcerations on upper endoscopy. Dry scooping refers to ingesting work out supplements without the recommended doses of solvent. This trend has been the subject of TikTok and other social media sites aimed at enhancing workout performance. While caustic ingestions leading to esophageal ulcers and strictures are well known, dry scooping ingestion of pre-workout powder as an etiology has not been described. Though caffeine may be the predominant content in such powders, the exact composition and ratios of other constituents, including amino acids, are less clear. Complete abstinence from ingestion of the pre-workout formulation and the use of a proton pump inhibitor therapy led to significant clinical and endoscopic recovery over a 4-week period. A thorough history of ingestions, including supplements, is critical when unraveling emerging etiologies of esophageal ulcerations.

Self-propelling thrombin powder enables hemostasis with no observable rebleeding or thrombosis over three days in a porcine model of upper gastrointestinal bleeding.

Hemostatic powders used to manage upper gastrointestinal bleeding continue to exhibit high rebleeding rates. Previously, self-propelling thrombin powder (SPTP) sprayed endoscopically managed severe Forrest Class 1A bleeds. Here, we evaluate SPTP in a 3-day recovery model of diffuse ulcerated bleeding. Five anesthetized pigs underwent an endoscopic mucosal snare resection to trigger a diffuse ulcer bleed and were treated with SPTP. The time to hemostasis and the amount of powder delivered were measured. Pigs were recovered and monitored. Pigs achieved hemostasis in 4.5 +/- 1.2 min (mean +/- SEM; n=5). At 3 days post-procedure, rescope showed no rebleeding in any animals. Measured blood parameters were not significantly different from baseline. There were no signs of foreign bodies or thromboembolism during gross necropsy and histopathology of key organs. SPTP is a promising novel material that stopped diffuse ulcer bleeds in 5 pigs without rebleeding or adverse local or systemic events.

Shaggy aorta-An autopsy analysis.

Shaggy aorta is defined as "very extensive atheromatous disease with diffuse ulcers associated with soft, loosely held debris and a paucity of actual thrombus" and often results in visceral or peripheral arterial embolization (shaggy aorta syndrome). Most of the studies are clinico-radiological with hardly any assessment of the pathological features. We present an autopsy analysis of shaggy aorta. A retrospective study of autopsied cases of shaggy aorta over 15 years was conducted. The involvement of the various segments of the aorta (ascending, transverse, thoracic, and abdominal) was correlated with the clinical manifestations and cardiac/extra-cardiac findings at autopsy. The mortality was categorized as those related to shaggy aorta (Group I), related to cardiac diseases (Group II), and those unrelated to cardiovascular diseases (Group III). Nil. In a span of 15 years, there were 76 cases of shaggy aorta affecting predominantly males (85.5%) and patients in the sixth decades of life (mean age of 64.5 years). The important associated cardiovascular risk factors included hypertension, tobacco use, and diabetes mellitus. Predominant involvement of the entire aorta and arch + descending aorta was seen in 39.5% and 35.5% of the cases, respectively. Regardless of extreme severity, only half of the patients (37 cases, 48. 7%) had clinical presentation due to shaggy aorta. The occurrence of shaggy aorta may be more common than expected, and it would be important to keep this possibility in mind even in asymptomatic elderly patients with cardiovascular risk factors since aorto-arterial manipulations and anti-coagulant therapy can prove detrimental in such patients.

STRONGYLOIDES STERCORALIS: A RARE CASE DIAGNOSED WITH ESOPHAGEAL SWAB SAMPLE

Strongyloides stercoralis is an opportunistic parasite that may cause severe and fatal disease in immunocompromised hosts. A 70-year-old man with a nonspecific history except pneumoconiosis had diffuse ulcers on the esophagus. Hence, only a swab sample could be performed. On microscopic examination of the swab sample, larvae and soils were observed in necrotic and inflammatory background. It was evaluated as compatible with S. stercoralis. At repeated endoscopic examination multiple biopsies were taken from the stomach. Larvae and soils were detected in crypt lumens with chronic inflammation in gastric mucosa. We presented this case with gastric involvement as a rare presentation that has not been reported in the literature before and was diagnosed by esophageal swab sample. Although infection is usually asymptomatic in the chronic phase, it carries a high mortality risk in immunocompromised hosts. So, it is important to scan the risk group. Swab sampling is an easy method for cytological examination.

A Case Study: Henoch-Schonlein Purpura in a Geriatric Patient with Gastrointestinal Involvement

IgA-mediated immune vasculitis known as Henoch-Schonlein purpura affects the small blood vessels in the joints, kidneys, GI tract, skin, and, less frequently, the brain and lungs. The present case report describes a 62-year-old female patient with Henoch-Schonlein purpura (HSP). HSP is a type of vasculitis that affects small vessels and is characterized by purpura, arthritis, abdominal pain, and kidney involvement. In this case, the patient presented with a rash with red lesions on the upper and lower limbs, crusted lesions on both lower limbs, and acute onset abdominal pain with hematochezia. Endoscopy revealed diffuse ulcerations in the stomach, duodenum, and right colon(fig.1). Biopsies revealed a leukocytoclastic vasculitis in the skin (fig2) and gastrointestinal tract. The patient had a history of hypertension and smoking, and laboratory investigations revealed leucocytosis, anaemia, and electrolyte imbalances. The diagnosis of HSP was made based on the clinical presentation, laboratory findings, and biopsy results. The patient was treated with a combination of steroids, antibiotics, and supportive care, which led to significant improvement of symptoms and laboratory findings. Steroid therapy was given according to guidelines for the treatment of HSP, and tapering the steroid therapy helped to improve gastrointestinal symptoms. The case report highlights the importance of careful monitoring and appropriate treatment of HSP in geriatric patients, as well as the potential benefits of immunosuppressant therapy.

A rare immunological disease, caspase 8 deficiency: case report and literature review

BackgroundCaspase-8 is a molecule in the FAS pathway that initiates apoptosis. One of the rarest autoimmune lymphoproliferative syndromes is caspase-8 deficiency. Immunodeficiency, splenomegaly, and lymphadenopathy are the common symptoms of this condition.Case PresentationA two-year-old boy entered this study with a fever of unknown origin (FUO) and dysentery. Moreover, he suffered from failure to thrive and was allergic to the cow's milk protein. His fever and dysentery did not respond to antibiotic therapy. The colonoscopy revealed diffuse ulcerations regions in the sigmoid along with skipped areas, mimicking Crohn's disease aphthous lesions. He represented very early-onset inflammatory bowel disease (IBD) and was diagnosed with the caspase-8 deficiency.ConclusionThere can be diarrhea or dysentery as the first or main symptoms of inborn errors of immunity (IEIs). The cause of diarrhea and dysentery in this case was early-onset IBD. One of the symptoms of IEIs such as caspase-8 deficiency is early-onset of IBD. Patients with early-onset had normal T cell count and low or normal immunoglobulin levels with insufficient immune response.

Polyarteritis nodosa: a case report of isolated large bowel involvement and surgical intervention

Polyarteritis nodosa (PAN) is a necrotizing vasculitis commonly affecting small-sized vessels and medium-sized vessels. We present the case of surgical resection for ischemic colitis secondary to PAN. A 26-year-old woman presented with left lower quadrant pain, diarrhea and bloody stools. Colonoscopy showed diffuse circumferential ulceration and necrotic lining from the sigmoid colon to the splenic flexure. A subsequent computed tomography angiography (CTA) revealed left colonic wall thickening, hypoenhancement and multiple inferior mesenteric artery aneurysms, concerning for vasculitis. Ensuing laboratory workup all returned negative. Worsening pain, left-sided peritonitis, tachycardia and rising white blood cell necessitated a colectomy from the proximal descending to upper rectum with an end colostomy. Surgical pathology showed features consistent with PAN. The patient improved significantly after surgery and was discharged on a steroid taper and later started on mycophenolate mofetil. This case is a unique presentation of isolated large intestinal involvement and was treated with both medical and surgical interventions.

Catastrophic Upper Gastrointestinal Tract Complications Following Corrosive Ingestion

Ingestion of caustic substances, either acidic or alkali, is a well-known cause of significant mortality and morbidity in the pediatric and adult populations. According to the pH, physical form, amount, and rate of ingestion of these substances, a wide range of immediate as well as possible subsequent chronic complications can result. Some patients may have minimal to mild symptoms. However, others can present with moderate or even serious symptoms, in the form of dysphagia, odynophagia, hoarseness, and epigastric pain. Herein we present a case of a 26-year-old male patient with a history of Nitric acid ingestion. He initially complained of severe chest and abdominal pain, associated with hematemesis, dysphagia, odynophagia, hypersalivation, and inability to tolerate oral intake. Initial Esophagogastroduodenoscopy (EGD) showed diffuse mucosal ulceration reaching the second part of the duodenum. Multiple subsequent EGDs revealed more extensive damage and subsequent esophageal stricture and pyloric stenosis for which multiple dilatation attempts were done. The patient received extensive supportive management during his hospitalization course, but the damage was severe enough that he was referred for surgical management. Such catastrophic sequels of caustic ingestion are still encountered, especially in developing countries. We present this case looking forward to raising awareness about this dangerous phenomenon and highlighting the significance of immediate identification and grading of the injury.

Iron-deficiency anemia following herpetic esophagitis in a previously healthy female

Herpetic Esophagitis is caused by the Herpes Simplex virus, which generally affects immunocompromised individuals and is rarely seen in healthy individuals. Symptoms are usually self-limiting. We report the case of a 68-year-old female who presented with odynophagia, dysphagia, and epigastric pain with no other underlying disease. Endoscopic findings of soft, nodular, and friable growths just above the squamocolumnar junction with diffuse ulcerations in the distal esophagus, led to the diagnosis. It was confirmed with a histopathological report which revealed multinucleated giant cells with eosinophilic intranuclear inclusions. During follow-up, laboratory investigations revealed iron deficiency anemia, which was the consequence of GI bleeding. Herpes Simplex virus esophagitis can occur in immunocompetent individuals and even it can cause food impaction and GI bleeding, which can lead to Iron deficiency anemia. Hence, follow-up of patients is important for early diagnosis and intervention of any complications that may arise.

S2190 A Case of Colorectal Signet Ring Cell Carcinoma Presenting as Ulcerative Recto-Sigmoiditis and Stricture

Introduction: Signet ring cell carcinoma accounts for about one percent of all colorectal cancers. It is an aggressive subtype of adenocarcinomas with the tendency for intramural spread and peritoneal carcinomatosis. Here, we reported a middle-aged male with circumferential colonic stenosis and inconclusive histology, found to have stage 4 colorectal signet ring cell carcinoma (SRCC). Case Description/Methods: A 41-year-old male without significant past medical history was referred to a gastroenterology clinic with bright red blood per rectum. Colonoscopy showed ulcerative recto-sigmoiditis with rectal bleeding, and there was stricture in the rectum, in the recto-sigmoid colon, and from anus to descending colon (Figure A). Biopsy was obtained from the stricture. The pathology revealed granulation tissue and abundant fibrinopurulent exudate showing small clusters, and individual atypical cells stained positive for CDX-2 immunostain. Unfortunately, the patient subsequently lost follow-up. Three months later, the patient was hospitalized for small bowel obstruction. CT showed markedly enlarged heterogeneous and edematous rectum, an abnormal mass within the posterior pelvis/rectum, retroperitoneal and pelvic lymphadenopathy with thickening and nodularity of the peritoneum. Biopsy was obtained from an inguinal lymph node with histological examination showing metastatic adenocarcinoma composed of poorly cohesive signet-ring cells (Figure B). Immunostains revealed that the neoplastic cells were strongly and diffusely positive for CDX2 and CK20 while negative for CK7, confirming a colorectal primary. Accordingly, the diagnosis of colorectal signet ring cell carcinoma was made. Discussion: The colonoscopic findings of colorectal SRCC could be nonspecific as diffuse circumferential thickening, stricture, or ulcerations. Typical pathological features may not appear on the initial biopsy sample. Immunohistochemical testing could help increase diagnostic yield and early identification of cancer cells. Our case hallmarked the importance of close follow-up for abnormal diffuse stricture and ulcerations in the colorectal area. These lesions may need to be rebiopsied, co-screened with abdominal imaging, and undergo an immunohistochemical investigation to characterize pathology further.Figure 1.: A: Colonoscopy: An intrinsic inflammatory circumferential fibrosis friable severe stenosis measuring 20cm (in length) * 9 mm (inner diameter) was found in the rectum, in the recto-sigmoid colon, and from anus to descending colon. B: Hematoxylin & eosin stain of tissue from lymph node showed metastatic adenocarcinoma composed of poorly cohesive signet-ring cells.

S2025 Eosinophilic Enterocolitis Improved After Breast Reduction

Introduction: Hypereosinophilic Syndrome (HES) is a group of disorders characterized by an accumulation of eosinophils within peripheral tissues or blood, with manifestations dependent on organ involvement. Eosinophilic gastrointestinal disorders (EGIDs) is one manifestation. In our case, it is believed that antigenic leakage from ruptured silicone breast implants was the proximate cause, as the patient had complete resolution of symptoms and normalization of labs after implant removal. Case Description/Methods: Our patient is a 59-year-old female with a history of asthma, who presents with new-onset sharp epigastric pain worsened with oral intake and associated with diarrhea. Recent evaluation at another hospital for leg weakness revealed peripheral eosinophilia and electromyography showed chronic inflammatory demyelinating neuropathy. After a full diagnostic evaluation for HES, she was diagnosed with idiopathic HES and treated with low dose steroids, IVIG and plasmapheresis without clinical improvement. At our hospital, labs revealed a white count of 35,000/μL, eosinophil count of 21,960/mm3 and IgE of 1299 IU/mL (Table). A CT scan revealed descending colon wall thickening, and intracapsular rupture of the right breast implant. Enteroscopy demonstrated erosions and ulcerations of the duodenum and jejunum. Colonoscopy showed areas of focal ulcerations with surrounding inflammation separated by normal mucosa and histology showed eosinophilic infiltration throughout. Video capsule endoscopy demonstrated diffuse inflammation and ulceration of the entire small bowel. She was empirically treated for strongyloides before starting high dose prednisone therapy. A month later, her breast implants were removed elsewhere. After a short interval, she was successfully tapered off steroids and achieved resolution of both her GI symptoms and lower extremity weakness, as well as normalization of lab work (Figure). Discussion: EGIDs are believed to be multifactorial with an emphasis on immunologic factors. Eosinophilic infiltration seen on biopsies and exclusion of secondary causes is required. Treatment is aimed at symptomatic improvement and removal of offending agent, as seen with our patient who experienced complete clinical resolution after removal of her silicone breast implants. Our case highlights a rare case of eosinophilic enterocolitis associated with a ruptured breast implant in an otherwise healthy patient and emphasizes the importance of remaining vigilant of all external factors that may be contributing to the disease process.Figure 1.: A, B: Small intestine duodenum & Jejunum. C, D: Colonic areas of inflammation. Table 1. - Initial labs versus labs post implant removal Units Reference Labs Initial Labs Labs post implant removal White Blood Count μL 4.0 - 10.5 35 x 103 7.1 Hemoglobin g/dL 11.2 - 15.7 9.6 11.2 Platelets μL 150-400 508 x 103 369 Eosinophil # μL 0.04 - 0.36 21,960 2.5 Eosinophils % 0.7-5.8 61 35 IgE IU/mL < 100 1299

S2704 A New Protein Losing Enteropathy: Autoimmune Cryptolytic Enterocolitis

Introduction: Protein losing enteropathy (PLE) can be classified into erosive, non-erosive, and increased interstitial pressure PLE. Presenting signs are often chronic non-bloody diarrhea, anasarca, abdominal pain, and weight loss. We present a complex case of a PLE presentation with a novel diagnosis. Case Description/Methods: A 30-year-old woman with a history of migraines presented with severe abdominal pain and chronic diarrhea. She was on long-term combined estrogen-progestin oral contraceptive pills (OCP) and chronic high dose non-steroidal anti-inflammatory (NSAID) medications. She was exposed to mycoplasma pneumoniae one month prior to presentation, without other infectious exposures. Laboratory tests were notable for hemoglobin 6 g/dL, albumin 0.5 g/dL, stool pathogen PCR negative, and fecal calprotectin >3000µg/g. Extensive infectious work up only revealed Mycoplasma IgM+. Inflammatory markers and alpha-1 anti-trypsin clearance were elevated, and she was found to have warm autoimmune hemolytic anemia. CT angiography excluded ischemia and vasculitis (autoimmune serologies were negative), revealing diffuse enterocolitis, confirmed on magnetic resonance enterography with ileal ulcers. Endoscopic evaluation revealed diffuse superficial ulcerations terminal ileum and colon with diffuse mucosal sloughing. Histopathology revealed diffusely injured crypts with crypt drop-out and minimal inflammation, without any findings to suggest infection, inflammatory bowel disease (IBD) or autoimmune enteritis (Figure). Normal B-cell switching studies excluded common variable immunodeficiency (CVID). Due to profound anasarca and inability to tolerate oral intake, albumin infusions and total parenteral nutrition were started. There was minimal response to systemic or topical steroids. Diarrhea was mildly improved with albumin repletion, and she was empirically started on vedolizumab. Discussion: Given the absence of classic IBD findings, negative infectious and immunological testing, we present the first case of autoimmune cryptolytic enterocolitis, a histopathologic diagnosis of unclear etiology and pathogenesis. The patient has responded to vedolizumab infusions as an empiric treatment for an IBD-like entity. Nevertheless, supporting the autoimmune component of this entity, is a possible concomitant potential pathway of molecular mimicry in the setting of post-mycoplasma infection along with chronic high dose NSAID and OCP exposure.Figure 1.: Terminal Ileum and Sigmoid Colon with Diffuse Crypt Dropout.

S3726 A Silent Gastric Adenocarcinoma With Peritoneal Carcinomatosis: A Case of a Newly Diagnosed Decompensated Cirrhosis Leading to a Fatal Diagnosis of a Silent Metastatic Malignancy

Introduction: Gastric carcinoma (GC) with peritoneal carcinomatosis is an advanced cancer with a median survival of less than 6 months. It is the third leading cause of cancer-related death. We present a case of an advanced Gastric Signet-ring cell carcinoma (GSRCC) with peritoneal carcinomatosis that presented as decompensated liver cirrhosis. Case Description/Methods: A 62-years-old African American male with HTN and DM type II presented with 4 week of worsening abdominal distension with pressure and worsening acid reflux. He denied alcohol, smoking and has no family history of gastrointestinal malignancy. Physical exam revealed a non tender but distended abdomen with dullness to percussion. CT scan of abdomen and pelvis (CT A/P) revealed liver cirrhosis and moderate ascites with mesenteric edema. EGD revealed small varices and poor distensibility of the stomach with diffuse mucosal congestion, granularity, and ulceration in the gastric body and antrum. Biopsy uncovered poorly differentiated GC with signet ring cells. Ascitic fluid cytology also confirmed adenocarcinoma with positive CK7 and CDX-2 markers. Repeat CT A/P with contract revealed nodular densities in the omentum with ascites. With the aforementioned test results, diagnosis of GSRCC stage P3 was made and he was started on palliative chemotherapy with mFOLFOX6 (modified folinic acid, fluorouracil, and oxaliplatin). He is doing well after 9 months of mFOLFOX6 initiation. Discussion: Most GC are diagnosed at advanced and incurable stage with a median survival of 6 months hence early diagnosis is key to increased survival. The only potential curative therapy is surgical resection for GC in situ whereas palliative chemotherapy is the probable therapeutic option for intraperitoneally-disseminated GC. Based on multiple retrospective studies, gastrectomy with systemic palliative chemotherapy did not significantly improve survival when compared to chemotherapy alone in intraperitoneally-disseminated GC. With rising in GSRCC cases, general population should be educated about its early signs and should seek medical attention sooner. Physicians likewise should have a low threshold for referring to gastroenterologist for endoscopy in a high-risk patient with any alarm symptoms or for persistent acid reflux or epigastric pain that is not relieved with a trial of PPI. Our patient had acid reflux for more than a year which was mildly controlled with antacid. Had he sought appropriate medical attention, his diagnosis could have been made at an earlier stage.

S3149 Resolution of Autoimmune Hepatitis With Concurrent Ulcerative Colitis After Colectomy: A Case Report

Introduction: UC is an idiopathic inflammatory disease results in diffuse inflammation and ulcers of the colonic mucosa. Hepatobiliary diseases are extraintestinal manifestations of UC. PSC is the most common condition; it shares a similar pathogenesis with IBD. However, some patients may develop AIH/overlapping syndrome, that may result from the chronic inflammation, alteration of the intestinal microbiota and mucosal barrier disruption resulting in toxins and bacterial translocation into the portal circulation. We aim to spot the light on a patient with severe UC complicated with AIH that resolved after colectomy. Case Description/Methods: A 37-year-old Caucasian female was diagnosed with UC at age 29, she had severe UC, colonoscopy showed pancolitis with extensive mucosal ulcerations proximal to the splenic flexure. LFTs have been persistently high during follow up with elevation of AST and ALT 7-8 times the upper limit of normal. Serology showed positive ANA, ASMA, and elevated IgG. Liver biopsy showed portal mononuclear cell infiltrates, nondestructive cholangitis, and mild fibrosis consistent with AIH. Due to persistent symptoms despite medical therapy, and long-standing disease, patient had a proctocolectomy with one stage ileal pouch-anal anastomosis. During a follow up period of 24 months after surgery, LFT has consistently trended down to normal, and the titer of ANA dropped to < 1:80. Discussion: Concurrent UC and AIH isn't commonly seen in practice. These patients have more tendency to develop the disease at younger age, severe disease, pancolitis, resistant to medical therapy, and higher risk of death or liver transplantation. Different pathogenesis behind this phenomenon have been suggested by researchers based on human or mice studies. Chronic inflammatory reaction, autoimmunity activation, and elevation of inflammatory markers could have resulted from the alteration of intestinal microbiota, disruption of the tight junctions proteins that increase the permeability of the colonic mucosa, resulting in toxins and bacterial translocation by the level of lipopolysaccharide to find their way to the liver through the portal circulation. Human studies showed that increased intestinal permeability correlated with the severity of the AIH. In our patient, we hypothesize that severity of the AIH has significantly improved after the patient had a colectomy in which the source of toxins has been eliminated. However, larger retrospective studies using the databases or prospective trials are suggested.

Characteristics of Children With Inflammatory Bowel Disease and Coexisting Celiac Disease Seropositivity.

Celiac disease (CeD) autoimmunity and coexisting inflammatory bowel disease (IBD) present a diagnostic dilemma. Our aims were to describe the phenotype of children with IBD and CeD seropositivity and evaluate provider confidence for diagnosing CeD in this population. We performed a single-center retrospective cohort study of subjects ≤18 years old with IBD and CeD seropositivity between 2006 and 2020. Subjects were considered to have IBD-CeD if they met CeD diagnosis by serology and histology per North American Society For Pediatric Gastroenterology, Hepatology and Nutrition guidelines and if providers suspected CeD as evaluated by a survey. The IBD-only cohort included seropositive participants that did not meet criteria for CeD. Demographic, histologic, gross endoscopic, and laboratory features were compared using Fisher exact test. Of 475 children with IBD, 8 had concomitant CeD, 5 had tissue transglutaminase (tTG) immunoglobulin A (IgA) > 10x upper limit of normal (ULN, P = 0.006), and 8 had villous atrophy (VA, P = 0.003) when compared with 17 seropositive participants with IBD-only. No children with IBD-CeD had esophageal eosinophilia, duodenal cryptitis, duodenal ulceration, or fecal calprotectin >250 µg/g. Factors that contributed to provider uncertainty for diagnosing CeD in IBD included the absence of VA and intraepithelial lymphocytes, the presence of neutrophilic and eosinophilic duodenitis, diffuse ulceration, elevated inflammatory markers, and immunosuppression therapy. Diagnosing CeD in children with IBD continues to be challenging. Although high titers of tTG IgA and VA increased provider confidence for diagnosing CeD in IBD, development of evidence-based guidelines are needed. They should better assess the importance of features atypical of concomitant CeD that contribute to uncertainty.

Molecular and oral manifestations of langerhans cell histiocytosis preceding acute myeloid leukemia

BackgroundLangerhans cell histiocytosis (LCH) is a heterogeneous neoplastic disorder that is rarely seen in patients aged 60 years and older. It is reported that elderly patients with LCH have a higher chance of having malignancies. In the oral cavity, patients with LCH can present with mucosal ulcers and extensive osteolysis, making it difficult for clinicians to make a proper diagnosis.Case presentationWe reported an 82-year-old Chinese woman with oral symptoms as the first presentation of LCH, and eventually developed acute myeloid leukemia (AML). She suffered diffuse ulcers involving the entire gingival mucosa and the left half hard palate, and had lost several teeth. Genomic DNA sequencing of the cells from LCH revealed multiple mutations in TET2, BRAF, SRSF2, NRAS, MAP2K4 and so on. The patient declined the BRAFV600E inhibitor (Vemurafenib). Although a dramatic improvement of the oral ulcers was achieved after symptomatic treatment, the patient developed acute myeloid leukemia (AML) and died.ConclusionsThis report presented the diagnostic difficulties of LCH with oral manifestations and highlighted the importance of radiological assessments and laboratory tests. Moreover, many of the mutations detected in our LCH patient are frequently seen in AML, suggesting that AML and LCH cells in this patient share the same origin.

Cutaneous lesions in a solid organ transplant recipient: A diagnostic dilemma.

We present the case of a 66-year-old female with a history of renal transplant in 1999 with new onset fevers and diffuse skin ulcerations. In this article, we present the diagnostic studies, differential diagnosis, and treatment decisions for the case.