bluuebunny/crossref_metadata_2025_split huggingface dataset:
2059 A Case of Hepatosplenic T-cell Lymphoma in a Patient on Vedolizumab for Crohn’s Disease
Authors: Fischer Monika , Walker Megan J. 
Published: October 2019

INTRODUCTION:
Hepatosplenic T-cell lymphoma (HSTCL) is a rare malignancy, most commonly affecting young males with often fatal prognosis. In patients with inflammatory bowel disease (IBD), HSTCL has been associated with thiopurine analog and tumor necrosis factor (TNF)-α inhibitor therapy; particularly when used in combination, the risk increases up to 4 fold. Development of HSTCL has not been reported in association with vedolizumab.

CASE DESCRIPTION/METHODS:
A 22-year-old male with severe, stricturing colonic Crohn’s disease of 10 years duration, on vedolizumab monotherapy every 4 weeks for 15 months, was incidentally noted to be pancytopenic in October 2016 and subsequently diagnosed with HSTCL by bone marrow biopsy. Previous IBD therapies included mesalamine, azathioprine, infliximab, adalimumab, intravenous immunoglobulin (IVIG), budesonide, certolizumab pegol and most recently 6 mercaptopurine (6-MP). The 6-MP was discontinued 1 year prior to HSTCL diagnosis. He achieved remission with chemotherapy, followed by autologous stem-cell transplant in September 2017. Unfortunately his disease relapsed only a few months later. He achieved a second remission with salvage chemotherapy, and proceeded to allogeneic bone marrow transplant in April 2018. Since diagnosis of HSTCL, he did not receive any therapy for Crohn’s disease and has been asymptomatic from his IBD. Colonoscopy demonstrated complete endoscopic and histologic remission as well. On last bone marrow biopsy, over 200 days post-transplant, there was no evidence of HSTCL relapse 24 months following HSTCL diagnosis.

DISCUSSION:
Fortunately, our case had a favorable outcome compared to those reported in the literature with a median overall survival of 10 months, and up to 17 months in patients who receive stem cell transplant. In addition, following stem cell transplant, our patient achieved cure of his Crohn’s disease. A recent review of patients with immunoregulatory disorders who developed HSTCL noted that 56% of patients were treated with TNF-α inhibitors versus 89% with immunomodulators, indicating that the latter pose higher risk. To our knowledge, there have not been reports of HSTCL associated with vedolizumab use nor is there a known mechanism by which vedolizumab could cause HSTCL. In our case, TNF-α inhibitor was stopped 5 years prior and thiopurine analog 1 year prior to diagnosis, raising the question as to how long the risk of HSTCL remains elevated after cessation of such agents.
2062 New Onset Colitis and Venous Thromboembolism in a Postpartum Adolescent
Authors: Rao Anita , Runde Joseph , Patton Tiffany 
Published: October 2019

INTRODUCTION:
We present a case of an adolescent female with a postpartum stroke in the setting of Disseminated Intravascular Coagulation (DIC) requiring therapeutic Heparin. Due to persistent hematochezia, she underwent upper and lower endoscopies which showed the presence of severely-active colitis. The patient was additionally found to have positive antiphospholipid antibodies, raising concern for Catastrophic Antiphospholipid Syndrome (CAPS). To our knowledge, this is the first reported case of presumed Inflammatory Bowel Disease (IBD) presenting initially in the immediate postpartum period and being associated with extensive venous thromboembolism (VTE).

CASE DESCRIPTION/METHODS:
Our patient is a 16-year-old G1P1 previously-healthy female with history of pre-eclampsia in the peripartum period who presented 9-days postpartum with complaints of right-sided weakness and inability to speak. She was found to have left cerebral vein thrombosis resulting in hemorrhagic stroke in the setting of DIC (Figure 1). This resulted in severe cerebral edema requiring hemicraniectomy, external ventricular drain placement, and subsequent ventriculo-peritoneal shunt placement. The patient was discharged to a chronic care facility but re-presented due to persistent hematochezia. Anticoagulation was held, and colonoscopy showed diffuse moderately to severely-active colitis in the rectosigmoid with the presence of pseudopolyps (Figure 2). Video capsule endoscopy showed a visually-normal small bowel with a severely-inflamed cecum. Her fecal calprotectin was elevated to > 1,000. She was started on IV Methylprednisolone as a bridge to eventual therapy with Adalimumab for presumed Ulcerative Colitis vs. Indeterminate Colitis. Further work-up showed laboratory-evidence of Antiphospholipid Antibody Syndrome, without laboratory-evidence of Systemic Lupus erythematosus (SLE).

DISCUSSION:
Patients with IBD are known to have an increased risk of VTE, and earlier studies have shown that IBD patients may have an even greater risk of VTE during pregnancy. However, this is the first reported case of IBD initially presenting in the immediate postpartum period and being associated with presumed Catastrophic Antiphospholipid Syndrome (CAPS), which is a life-threatening disease due to multi-organ thrombosis. Our patient showed a very positive clinical and histological response to Adalimumab therapy, with cessation of hematochezia. This further strengthens the presumed diagnosis of IBD instead of ischemic colitis.
2064 Favorable Outcomes Among IBD Patients Undergoing Bariatric Surgery: A Case Series
Authors: Stanfield Dylan , Spiel Andrew , Saha Sumona 
Published: October 2019

INTRODUCTION:
The prevalence of obesity continues to rise worldwide. It has been demonstrated that conditions such as T2DM and gallbladder disease, among other comorbidities, are related to Class II and III obesity (BMI ≥ 35). To combat this issue, bariatric surgery has been shown to be the most effective and durable weight loss method in individuals with a BMI ≥ 40 or BMI ≥ 35 with an obesity-related comorbidity. The rate of obesity among IBD patients parallels that of the general population and is approaching 30% nationwide. In recent years, there has been an increase in clinical research supporting the use of bariatric surgery within the IBD population as a safe and efficacious treatment option.

CASE DESCRIPTION/METHODS:
We present a case series of patients with a pre-operative diagnosis of IBD who underwent bariatric surgery at our hospital. Selection process was based on an ICD 9 or 10 diagnosis of IBD prior to Bariatric Surgery referral. Patients with a diagnosis of IBD but not followed by a gastroenterologist were excluded from this review. Patient data was extracted by the Clinical Research Data Service team from the electronic medical record. They identified 144 patients with IBD diagnoses who were seen at bariatric surgery clinic from 2003-2018. 5 IBD patients (3 CD, 2 UC) who underwent bariatric surgery were included in this data set and analyzed for IBD disease activity, operative complications, mortality, post-operative complications and overall excess weight loss (%EWL) at 6-month follow-up. All IBD patients who had bariatric surgery at this facility were included in data analyses. Bariatric surgical procedures included sleeve gastrectomy and Roux-en-Y. Mean age at time of referral was 43 years ± 12. One major perioperative complication was identified in a UC patient, pulmonary embolism complicated by splenic infarct. %EWL at 6-month follow-up was 35.4% ± 12.8. No patients required rehospitalization and mean length of hospital stay for the operation was 2.5 days ± 1.7.

DISCUSSION:
There were no IBD-related complications (glucocorticoid use or rehospitalization) seen within 30 days of surgery and no major complications (including SBO) were noted within a median follow-up of 54 months. As clinical data continues to emerge regarding bariatric surgery in the obese IBD population, it has become increasingly apparent that the pre-operative diagnosis of IBD should not exclude considerations for utilizing a surgical treatment modality for weight reduction in select patients.
2069 Colocolonic Intussusception in a Patient With Ulcerative Colitis
Authors: Patel Zinal M. , Ali Mohammad F. , Das Kanak , Friedel David , Maghsoodi-Deerwester Mona , Gupta Mala , Stavropoulos Stavros 
Published: October 2019

INTRODUCTION:
Intussusception is common in children, but rare in adults accounting for 5-16% of all cases of intussusception. Giant pseudopolyps are polyps that measure more than 1.5 cm in diameter. These can function as lead points for intussusception in patients with inflammatory bowel disease (IBD). We present here an unusual case of intussusception due to a giant pseudopolyp in an adult patient with ulcerative colitis. There are only a limited number of case reports that describe this occurrence.

CASE DESCRIPTION/METHODS:
A 20-year-old man with a history of ulcerative colitis in remission on 5-ASA and 6-Mercaptupurine was found on surveillance colonoscopy to have a large inflammatory pseudopolyp resulting in partial obstruction. He was asymptomatic and physical examination was unremarkable. CT Abdomen/Pelvis showed transverse colon intussusception (Figure 1). He underwent a repeat colonoscopy with plan for endoscopic submucosal dissection of the large polyp. A 15 × 15 cm, frond-like/villous, broad based pseudopolyp without a stalk was seen in the proximal transverse colon (Figure 2a). Due to the large size of the polyp and inability to visualize the dissection plane, endoscopic submucosal dissection could not be performed. Debulking was achieved using piecemeal mucosal resection with a snare. A 15 × 15 cm area was resected and retrieved (Figure 2b). Histopathology showed fragmented pseudopolyp with ulceration, extensive granulation tissue and regenerative epithelial changes (Figure 3). He did well post procedure and was discharged home. He was advised to undergo colectomy but he remains undecided. He will undergo surveillance colonoscopy in 6 months.

DISCUSSION:
In adults, intussusception is a rare finding and most cases are due to colonic malignancy. Colocolonic intussusception secondary to giant pseudopolyps is an extremely rare occurrence in IBD patients. Presentation of these patients can vary from asymptomatic, abdominal pain, rectal bleeding, obstruction or intussusception. Although giant pseudopolyps do not have a malignancy risk, there has been one reported case of occult malignancy in a giant pseudopolyp. In addition, IBD patients at baseline are at an increased risk for malignancy. Asymptomatic patients can be managed medically. Successful endoscopic therapy with polypectomy has been reported in a few cases of giant pseudopolyps. Complications such as obstruction or intussusception require surgery. However, all of these patients will require frequent endoscopic surveillance.
2070 Sweet's Syndrome as the Initial Presentation of Crohn's Disease
Authors: Ryan-Fisher Courtenay , Siddique Osama , Birk John 
Published: October 2019

INTRODUCTION:
The diagnostic criteria for Sweet's syndrome include fever, peripheral neutrophilia, erythematous skin lesions and diffuse neutrophilic dermal infiltrate. Classically, the lesions and symptoms promptly improve with the initiation of systemic corticosteroids. This is a rare case of Sweet’s syndrome as the initial presentation of Crohn’s disease.

CASE DESCRIPTION/METHODS:
A 23-year-old African-American male with no past medical history presented to our ED with fever and rash. He reported a sudden onset mildly sensitive rash on his bilateral lower extremities, preceded by two weeks of fever, myalgia and headaches. He denied arthralgia but endorsed recent weight loss and sporadic aphthous ulcers. He developed watery, non-bloody diarrhea in the ED. His abdomen was soft and non-tender. Examination of the rash revealed erythematous nodules, some studded with pustules and a few ulcerated nodules. He was febrile to 102.7 ºF with tachycardia. Labs were significant for leukocytosis at 22.5 (68% neutrophils), thrombocytosis at 515, microcytic anemia (Hb 9.6), elevated inflammatory markers (CRP 326 mg/L; ESR >130 mm) and AST:ALT 64:41 U/L. Broad spectrum antibiotics were started due to concerns for sepsis. Blood, wound, and stool cultures, including Clostridium difficile were all negative. Fecal calprotectin was 32. His immunological workup showed ANA, RF and anti-GBM negative; normal IgM, IgG, IgA, C3 and C4. HIV, Hepatitis B and C and QuantiFERON were also negative. ANCA was positive at 1:320, with positive PR3. CT scan of the abdomen showed proctitis and ascending colitis with no lymphadenopathy. Colonoscopy showed mild-moderate inflammation of the left sided colon with skip lesions in the transverse colon, without terminal ileum involvement, with biopsies showing focal acute cryptitis and rare crypt abscesses. Skin biopsy revealed neutrophilic dermatitis and panniculitis without vasculitis. Once infection was ruled out, prednisone 60 mg was started and the rash and fever rapidly improved.

DISCUSSION:
Sweet’s syndrome is considered part of the family of neutrophilic dermatoses, which also includes pyoderma gangrenosum, and is considered an unusual extra-intestinal manifestation of IBD. Sweet's syndrome is also associated with malignancies, infections, pregnancy and medications. The temporal relationship between Sweet's syndrome and IBD activity is variable, but the majority of cases present after the diagnosis of IBD, with few cases in the literature with Sweet’s syndrome as the presenting symptom for IBD.
2073 A Rare Case of S. Constellatus Bacteremia Secondary to a Perianal Abscess in an Individual With Crohn's Disease
Authors: Gallagher Lauren T. , Dayah Rawan , Qureshi Sidra 
Published: October 2019

INTRODUCTION:
Organisms of the Streptococcus anginosus group consists of three distinct streptococcal species: Streptococcus intermedius, Streptococcus constellatus, and Streptococcus anginosus. These organisms are commonly associated with abscesses, but polymicrobial infections in the setting of an immunocompromised patient can infrequently cause bacteremia. Here we report the case of a 37-year-old male with Crohn’s disease who was found to have bacteremia secondary to a perianal abscess due to a co-infection of Streptococcus constellatus and Parvimonas micra.

CASE DESCRIPTION/METHODS:
A 37-year-old male with Crohn’s disease who presented with fever, leukocytosis and rectal pain was found to have bacteremia due to a polymicrobial infection of Streptococcus constellatus and Parvimonas micra. A CT abdomen and pelvis performed excluded any acute abdominopelvic disease. A MRI was done to determine a source of infection which showed a left perianal abscess measuring 3.2 × 1.6 × 4.7 cm. Our patient underwent an examination under anesthesia with biopsy, anoscopy and surgical drainage with seton placement. He received intravenous antibiotic therapy with Ceftriaxone and Metronidazole for three days and was discharged on a two week course of oral Augmentin after two repeat blood cultures were negative. Patient continues to have the seton drain in place six months from surgery with no complications or reoccurrence of infection.

DISCUSSION:
This case report sheds light on a rarely described pathogen causing bacteremia and provides a comprehensive approach to diagnosis and management. S. constellatus is a common cause of abscesses in the alimentary tract but rarely causes infection in blood. Co-infection with an anaerobe in the setting of an immunocompromised individual such as Crohn's disease increases the likelihood for blood infections. Bacteremia from this group must prompt evaluation for possible intra-abdominal abscess, deep-seated head and neck infection, brain abscess and endocarditis to determine the underlying source of infection. Treatment of infections due to the S. anginosus group consist of abscess drainage and antimicrobial therapy with Ceftriaxone being the preferred agent.
2074 Small Bowel and Colonic Ulcerations in a 19-Year-Old Male With Disseminated Adenovirus: An Atypical Presentation of Inflammatory Bowel Disease?
Authors: Chen Brendan , Bakhit Mena , Fine Sean 
Published: October 2019

INTRODUCTION:
Enteritis and colitis have wide differential diagnoses including inflammatory bowel disease, infections, and ischemia. We present an unusual case of an 19-year-old male who presented to the intensive care unit with disseminated adenovirus and required a prolonged hospital course who went on to develop ongoing symptoms of non-bloody high output diarrhea. A colonoscopy was performed and demonstrated significant right sided colonic disease with ulcerations in a patchy distribution as well as small bowel inflammation of unclear etiology, however given the appearance was likely felt to be a new presentation of Crohn’s disease.

CASE DESCRIPTION/METHODS:
A 19-year-old male with a history of childhood asthma presented with 1 week of fatigue, cough, and sore throat. Patient was admitted to the intensive care unit for septic shock and acute respiratory distress syndrome secondary to disseminated adenovirus, requiring intubation and ultimately extracorporeal membrane oxygenation. Patient’s course was complicated by failure of enteral feeds requiring total parenteral nutrition and acute renal failure requiring CVVH and hemodialysis. On hospital day 14 patient began having non-bloody high output diarrhea. C. difficile toxin and stool cultures were negative. Fecal calprotectin was elevated to 681 mg/kg. A colonoscopy was performed revealing diffuse inflammation with linear erosions, mucus and deep ulcerations in the terminal ileum in addition to patchy inflammation characterized by erythema, cobblestoning, and deep ulcerations from the transverse colon to the cecum, all concerning for Crohn’s disease versus viral etiologies. Patient was empirically started on steroids with significant improvement of diarrhea. Pathology showed acute neutrophilic inflammation and chronic lymphohistiocytic inflammation of the mucosa without granulomas with the differential diagnosis felt to be viral enteritis or atypical inflammatory bowel disease.

DISCUSSION:
Crohn’s disease is diagnosed by clinical, endoscopic, and pathologic findings. However, the pathogenesis of IBD remains unclear. The two-hit hypothesis, where genetic predisposition and environmental factors cause a cascade pathway to IBD has been proposed as a likely mechanism. Factors including pathogens and diet, like adenovirus infection and TPN in our case, may be environmental triggers for IBD. In addition, this case highlights the need to always consider IBD in an acutely ill person even when treatments such as antibiotics and TPN may be other culprits for diarrhea.
2075 Tandem Therapy Using Cyclosporine and Tofacitinib for the Treatment of Severe Ulcerative Colitis
Authors: Cleveland Noa Krugliak , Masching Alexandra , Rubin David T. 
Published: October 2019

INTRODUCTION:
Although there are evolving options for the treatment of ulcerative colitis (UC), the timing and sequencing of newer treatments has not been sufficiently studied. Tofacitinib (tofa) is a non-selective Janus kinase inhibitor that is now available for patients with moderately to severely active UC, but its use in combination with other therapies has been limited due to trial design, labeling and evolving experience in these patients. We present a patient with medically refractory severe UC who was successfully treated with cyclosporine and then maintained with tofa.

CASE DESCRIPTION/METHODS:
A 24-year-old man with a history of primary sclerosing cholangitis (PSC) and overlap auto-immune hepatitis (AIH) was diagnosed with ulcerative pancolitis at the age of 20. He was maintained in clinical remission for two years with mesalamine. A subsequent clinical relapse required steroid therapy, then vedolizumab loading and q8w maintenance, but due to incomplete response, he was escalated to monthly infusions and after clarification of his overlapping liver disease, azathioprine was added. Despite these treatments, he was symptomatic, steroid-dependent, and had persistent evidence of endoscopic inflammation, CRP 29 mg/L and albumin 3.3 g/dL. Infliximab (IFX) 10 mg/kg loading and q8w dosing was initiated, but he was still steroid-dependent despite IFX trough 11 μg/mL. IFX was increased to 10 mg/kg q4w. Due to lack of response, he was admitted for IV cyclosporine (CsA). CsA at 3 mg/kg continuous infusion was initiated and titrated to a target serum level of 300-400 ng/ml. By day 6 he was in near clinical remission and discharged on oral CsA 200 mg BID. He was then transitioned from prednisone to ileal-release budesonide (for his liver disease). After two months of sustained clinical remission we stopped the CsA for 48 hrs and then initiated tofa at 10 mg PO twice a day. At 17-month follow-up he remains in clinical remission receiving tofa 10 mg BID and budesonide 6 mg daily, and has a normal CRP and normal liver enzymes.

DISCUSSION:
This is a patient with medically resistant UC, PSC and AIH successfully induced into deep remission using CsA and maintained with tofa (and budesonide for his liver disease, azathioprine should not be used with tofa). The half-life of cyclosporine (8.5 hrs) allowed for short washout period prior to initiation of tofa, and therefore prevented the risk of overlapping immune suppression and toxicity. This is the first report of such a combination therapy approach.
2077 A Rare Case of Migratory Urticaria Causing Dysphagia During an Acute Flare of Ulcerative Colitis
Authors: Alexis Jamil O. , Qiu He , Shen Janice G. , Sultan Keith 
Published: October 2019

INTRODUCTION:
Ulcerative colitis (UC) is characterized by chronic mucosal inflammation limited to the colon. The most common manifestation of UC is progressively worsening hematochezia that may be accompanied by extraintestinal manifestations including, peripheral arthritis, uveitis, primary sclerosing cholangitis, fatty liver, venous/arterial thromboembolism, and pulmonary disease. Erythema nodosum (EN) and pyoderma gangrenosum (PG) are the most common dermatologic manifestations. Uncommonly, UC may present with unpredictable urticaria and angioedema that may be life threatening. We present a rare care of acute UC flare with migratory urticaria as the initial presenting symptom.

CASE DESCRIPTION/METHODS:
A 25-year-old woman with UC presented with 1 week of abdominal pain, bloody stools, bilateral ankle and knee pain with non-blanching rash over her right shoulder, left armpit, and left groin. GI PCR and C. difficile assay were negative. Rheumatoid factor, double stranded DNA, anti-cyclic citrullinated peptide antibodies, anti-Smith antibody, anti-ribonuclear protein, and C1 esterase inhibitor were negative. The patient was started on cetirizine with resolution of the arthralgia. However, new rashes were noted under right axilla and left antecubital fossa over a two day period. Patient subsequently developed dysphagia and left sided mandibular swelling. Indirect laryngoscopy was unremarkable. She was immediately treated with IV methylprednisolone and transitioned to a slow oral steroid taper over 3 months with improvement of gastrointestinal and dermatologic symptoms. Skin biopsies revealed superficial and mid-dermal perivascular lymphocytic infiltrate consistent with urticaria.

DISCUSSION:
This patient presented with unpredictable migratory skin lesions involving multiple mucosal surfaces that brought concerns of possible airway involvement. The etiology was initially unclear as limited infectious and rheumatologic work up was negative, as well as negative allergen and medication exposure. Cutaneous vasculitis was considered, however biopsy showed urticaria. Patient’s symptoms significantly improved after the initiation of oral steroids. Migratory urticaria with associated angioedema can be a manifestation of an acute flare of UC that warrants evaluation by otolaryngologist and prompt initiation of steroids. Glucocorticoids may address symptoms of a pro-inflammatory state while allowing for more time to rule out other etiologies of extraintestinal manifestations of a patient’s presentations.