bluuebunny/crossref_metadata_2025 huggingface dataset:
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.
2079 Coexistence of Lymphocytic Colitis in a Patient With Ulcerative Colitis
Authors: Sadiq Omar , Alimirah Maryam , Kanduru Chakravarthy
Published: October 2019

INTRODUCTION:

Microscopic colitis (MC) is an inflammatory condition of the colon that is histologically divided into two different conditions, lymphocytic colitis (LC) and collagenous colitis. Initially suspected to be unrelated to inflammatory bowel disease, a growing body of evidence now suggests a common pathophysiology between MC and IBD. We describe a case report of a patient presenting with both LC and UC.

CASE DESCRIPTION/METHODS:

A 59-year-old Caucasian, nonsmoking female with a history of diabetes underwent a colonoscopy for evaluation of constipation which demonstrated significant colitis, consistent with UC. Her disease was managed with mesalamine however due to social restrictions, clinical follow up and management of her disease was limited. As part of age appropriate management, patient underwent routine screening colonoscopy 15 years after her initial diagnosis as an open access procedure. During the procedure patient was noted to have terminal ileum apthae with Mayo score 2 pancolitis. Random biopsies taken during the procedure demonstrated focal acute inflammation. In follow up clinic visit, patient endorsed having 3-4 non-bloody, loose bowel movements daily but was otherwise asymptomatic. She was started on balsalazide capsules with improvement in stool consistency and reduction in frequency of bowel movements. Shortly after starting balsalazide patient lost insurance access and was unable to follow up as directed. Patient returned to gastroenterology clinic two years later endorsing 4-5 non-bloody, loose bowel movements every day. Fecal calprotectin at this time was noted to be 26.7 mg/kg (normal < 50 mg/kg) however due to ongoing symptoms balsalazide was restarted. Repeat colonoscopy was performed to evaluate for dysplasia and ongoing symptoms which demonstrated Mayo score 1 pancolitis. Random biopsies taken during the procedure demonstrated chronic active colitis with superimposed lymphocytic colitis. Patient was started on budesonide in addition to balsalazide with reduction in stool frequency and improvement in consistency.

DISCUSSION:

In literature, coexistence of UC and LC is described in 12 cases, typically in patients with complete remission of UC. Here we describe a case of active UC further complicated by lymphocytic colitis which was successfully managed with a combination of a 5-aminosalicyclic-acid derivative and glucocorticoids. This case emphasizes the importance of looking for alternative causes of symptoms in patients with mild colonoscopic findings.
2090 EBV Colitis With Fulminate Ulcerative Colitis: A Clinical Dilemma
Authors: Lindsey Adrian , Ahmad Dina , Kurin Michael , Sinh Preetika , Lisgaris Michelle , Rodriguez Benigno , Nguyen David , Charles Ronald , Farag Rosemary
Published: October 2019

INTRODUCTION:

Epstein-Barr virus (EBV) is a virus of the herpes family. EBV can be detected in the gastrointestinal tissue of patients with inflammatory bowel disease(IBD). However, the clinical significance of EBV colitis in ulcerative colitis (UC) is unclear. We present an interesting and rare case of EBV colitis in a patient with fulminant UC.

CASE DESCRIPTION/METHODS:

A 73-year-old male with history of hypertension and recent diagnosis of UC who was doing well on mesalamine developed a flare with acute onset diarrhea, abdominal pain, and rectal bleeding. Stool tests were negative for C difficile and common bacterial and viral pathogens. Fecal calprotectin was >2000 μg/mg, and CT abdomen/pelvis was notable for thickened colon walls. Colonoscopy showed moderate inflammation with aphthous ulcerations throughout the colon with sparing of the cecum. Patient was started on oral steroids and transitioned to adalimumab. He continued to have weight loss and diarrhea and was switched to vedolizumab after two months. Due to continued symptoms, repeat colonoscopy 6 months later showed severe inflammation with confluent ulcerations in continuous and circumferential pattern from rectum to transverse colon (Figure 1). Laboratory workup was notable for anemia (hgb 9.0 g/dL), hypoalbuminemia (2.9 g/dL) and high C-reactive protein (3.22 mg/dL). Patient was admitted for IV steroids. Infliximab was started on day 5 due to no improvement in symptoms. At this time biopsies obtained during colonoscopy were reported to be positive for EBV-encoded small RNA by in-situ hybridization (Figure 2). EBV was also detected in whole blood by PCR. IV gancyclovir was started. After 7 days of treatment with gancyclovir, repeat flexible sigmoidoscopy showed no significant improvement. Immunostains for EBV, CMV, and HSV were negative. EBV PCR declined (60,000 to 30,000 IU/ml). A subtotal abdominal colectomy with end ileostomy and rectal stump was eventually performed without any complications. Patient gained weight and did well post operatively.

DISCUSSION:

EBV colitis with fulminant UC is rare. The clinical implication of EBV colitis and treatment benefits are unclear. In addition, there is lack of a standard therapy for EBV infection with case reports showing improvement with gancyclovir. Our case of true EBV colitis with fulminant UC illustrates the importance of considering a superimposed infection in patients with IBD refractory to steroids and immunosuppressive therapy early in the course of the disease to prevent progression.
2091 Orofacial Granulomatosis: The Proof Is in the (Cinnamon) Pudding
Authors: Crooks Benjamin J. , Limdi Jimmy K.
Published: October 2019

INTRODUCTION:

Orofacial granulomatosis (OFG) can present in isolation or in conjunction with other systemic disease including Crohn’s disease (CD). Data suggests that CD presenting with OFG may represent a distinct sub-phenotype of CD with more extensive inflammation and perianal disease [1]. Treatment of the oral manifestations of CD is typically challenging with numerous strategies proposed including topical and intralesional agents, immunomodulators, biological agents and dietary interventions.

CASE DESCRIPTION/METHODS:

A 24-year-old female with CD affecting the upper gastrointestinal tract, small and large bowel and perianal disease was referred to our unit for further management. She was diagnosed at the age of 19 when she had an emergency admission with jejunal obstruction. A jejunal resection with primary anastomosis was performed. She was managed with Infliximab and Azathioprine. She developed thickening of her buccal mucosa (Figure 1). A biopsy confirmed chronic inflammation with several non-necrotic granulomas in keeping with OFG. The patient was commenced on prednisolone with some improvement of the oral swelling however it later recurred requiring further oral steroids. Despite pro-active optimisation of Infliximab and Azathioprine levels, her oral manifestations of Crohn’s disease worsen (Figure 2). A cinnamon- and benzoate-free diet was recommended in liaison with specialist IBD dietitians with a dramatic improvement of her oral swelling within 4 weeks and near resolution in 8 weeks (Figure 3). Following 12 months of the diet the patient was advised to start re-introducing cinnamon and benzoates gradually into her diet and since this there has been no recurrence of oral symptoms. She has been maintained on an anti-TNF and Azathioprine for treatment of her ileo-colonic and perianal disease.

DISCUSSION:

There is good evidence for the efficacy of a cinnamon- and benzoate-free diet in patients with OFG raising the possibility of a dietary antigen and subsequent hypersensitivity in its aetiology. In our case the excellent response of the OFG to this diet, following failure to respond to conventional treatment for CD, is of interest. The debate regarding whether OFG is a distinct disease entity from oral CD continues. More studies are required to guide treatment options in this group of patients.
2093 Uncharted Territory: Biologic Therapies for Elderly Patients With Refractory Ulcerative Colitis
Authors: Figueredo Carlos , Boroda Konstantin , Hertan Hilary
Published: October 2019

INTRODUCTION:

Ulcerative colitis (UC) is becoming more common among people older than 65. However, diagnosis in these patients is challenging due to the presence of comorbidities that can resemble UC and symptoms being more subtle. UC treatment guidelines are based on trials of short duration that often exclude elderly patients. Therefore, when treating this population, choice of therapy should be tailored for each patient individually. The use of biologic agents in this population is controversial given the lack of strong evidence-based trials; with most data coming from the rheumatology literature. It can also be difficult as benefits are masked by multiple comorbidities and long term progression of the disease. We present a case series of two patients with refractory UC who had good outcomes with biologic therapy.

CASE DESCRIPTION/METHODS:

Case 1: 87-year-old male with left sided UC and history of prostate cancer treated with brachytherapy in 2013, complicated by radiation proctitis. He was initially maintained on 5-ASA, but had numerous hospitalizations with persistent diarrhea, rectal bleeding, dehydration, and C. difficile colitis over the course of 1 year. Sigmoidoscopy revealed severe “Mayo 3” colitis (Figures 1 and 2). He became steroid dependent, so was then started on adalimumab with improvement of his Disease Activity Index and no further UC related hospitalizations. Case 2: 80-year-old female with severe UC pancolitis diagnosed in 2017 and comorbidities of HTN, NIDMM, HLD, asthma, GERD, and stroke. She was initially on mesalamine enemas, which she did not tolerate due to a salicylate allergy. She was then switched to hydrocortisone enemas, then switched to adalimumab, with resultant persistent recurrence of flares. She is now on infliximab and in remission since 2018.

DISCUSSION:

The efficacy of biologic agents in the elderly UC patients, as compared to younger patients, has not been studied extensively. However, our experience has shown good outcomes in this population. Possible adverse effects of anti-TNF therapy include higher rates of cardiovascular diseases (e.g, heart failure), malignancy, psoriasis, and opportunistic infections. Advantages of this therapy are low rates of drug interactions and lesser impact on comorbidities when compared with usual treatments like steroids or 6-MP. Our experience supports the use of biologics in elderly patients with refractory UC.
2094 Segmental Colitis Associated With Diverticulosis Progressing to Ulcerative Colitis: An Under-Reported Occurrence
Authors: Omer Anam , Pinillios Hugo L.
Published: October 2019

INTRODUCTION:

Segmental colitis associated with diverticulosis (SCAD) is chronic inflammation of the interdiverticular mucosa, usually involving the sigmoid colon. This entity may mimic ulcerative colitis (UC) in its clinical manifestations and endoscopic appearance, however unlike UC, the rectum is usually spared. We report a rare case of SCAD progressing UC.

CASE DESCRIPTION/METHODS:

A 59-year-old female was referred for an outpatient colonoscopy for evaluation of a two-month history of hematochezia and diarrhea. Past medical history was significant for gastroesophageal reflux disease with esophagitis. Physical examination revealed no tenderness in the abdomen and normal bowel sounds. Rectal exam revealed small non-bleeding external hemorrhoids. Laboratory examination was significant for a low hemoglobin of 11.4 g/dL. Colonoscopy revealed patchy loss of vascular pattern, edema and hyperemia in the sigmoid colon associated with several large diverticula. Rest of the colon including the rectum and terminal ileum was unremarkable. Histologic evaluation of the specimens taken from the inflamed sigmoid colon showed expansion of the lamina propria with moderate chronic inflammation. No acute inflammation including acute cystitis or crypt abscess was present. The patient was diagnosed with SCAD and prescribed a two-week course of antibiotics with partial improvement in her symptoms. The patient was lost to follow-up for two years and presented again with hematochezia and iron deficiency anemia. A repeat colonoscopy revealed mild rectal erythema and an interval increase in inflammation of the sigmoid colon typical for UC. Several pseudopolyps were also seen. Histologic examination of the specimens taken from the sigmoid colon and rectum showed severe chronic active colitis with crypt architectural distortion compatible with UC. Crypt architectural distortion was also noted in the remaining endoscopically normal colon.

DISCUSSION:

The pathogenesis of SCAD remains unclear, however, mucosa prolapse, fecal stasis and localized ischemia is frequently implicated. Even though SCAD is recognized an independent disease progress, there have been reports of cases of SCAD progressing to inflammatory bowel disease (IBD). A high index of suspicion should be kept for patients with SCAD who do not respond to the main stray of treatment with antibiotics, anti-inflammatory agents and steroids. Also, patients with SCAD should be considered for close follow-up and surveillance endoscopy as this disease may co-exist or progress to IBD.
2095 Hemolytic Uremic Syndrome in a Young Female, Later Diagnosed With Crohn's Disease
Authors: Neupane Rabin , Kohli Akshay
Published: October 2019

INTRODUCTION:

Hemolytic uremic syndrome (HUS) is defined by microangiopathic hemolytic anemia, thrombocytopenia, and acute kidney injury. HUS has mostly been associated with Escherichia Coli infection but also seen with systemic infections, pregnancy, rheumatic disease and malignancies. Although, HUS has been described in previously diagnosed case of crohn’s, concomitant occurrence of both is a rarity. We describe a case of 37-year-old female, with acute onset of HUS, who was subsequently diagnosed with crohn's.

CASE DESCRIPTION/METHODS:

32-year-old female presented with complaints of abdominal pain, and bloody diarrhea for 5 days, which persisted despite antibiotic therapy. In ED, laboratory tests showed anemia, thrombocytopenia, AKI and Lactic acidosis. Her hemoglobin dropped to 6.7 g/dl and platelets went down to 48k. LDH was elevated and haptoglobin was low. Peripheral smear showed moderate schistocytes. However, shiga toxin was negative. Patient was managed symptomatically for HUS but her symptoms persisted. Imaging revealed thick-walled transverse and left colon. Colonoscopy showed mucosal ulceration and severe colitis with shallow and deep ulcers in ascending, transverse, and descending colon but rectum was normal. Push enteroscopy revealed a small esophageal ulcer. Biopsy showed granulomatous inflammation suggestive of Crohn's. Hence the patient was started on sulfasalazine. Patient had significant clinical recovery within a month and repeat colonoscopy showed healing congested mucosa.

DISCUSSION:

Although HUS is predominantly described in the pediatric population, its incidence is rising in adult population. It has been found to involve the transverse and ascending colon, much like Crohn's disease. A few reported cases has showed interchanging clinical features between HUS and IBD. Cumulative occurrence of the both was considered rare, however, cases of HUS in patient who were already diagnosed with Crohn's has been seen. In this case, the patient presented with the clinical features of HUS as her first and only manifestation of her Crohn's. Rapid recovery and resolution of symptoms within four weeks of starting the appropriate treatment presents the importance of the appropriate management on the clinical recovery. Hence, with the growing incidence of concurrent occurrence of both entities, early colonoscopy with biopsy can be beneficial in cases of HUS not responding to treatment, even if the patient has no history of IBD, as this could be their first presentation.
2097 A Double Whammy: A Case of Anterior Uveitis and Optic Neuritis in Patient With Crohn’s Disease on TNF Inhibitor Therapy
Authors: Kang William D. , Dominguez Cristian , Sayana Hari
Published: October 2019

INTRODUCTION:

Crohn’s disease (CD) is characterized by chronic transmural inflammation which can be managed with TNF inhibitor therapy. These medications are overall safe but can have uncommon yet significant side effects. This is a case of anterior uveitis with underlying optic neuritis secondary to chronic anti-TNF therapy.

CASE DESCRIPTION/METHODS:

A 53-year-old male with a history of Crohn’s colitis diagnosed in 1997, presented to ophthalmology with bilateral decreased visual acuity. He had failed multiple therapies for CD in the past, including Infliximab, Certoluzimab, azathioprine, and Methotrexate. He was on clinical remission on Adalimumab and oral mesalamine. Prior to therapy 18 months ago, his most recent colonoscopy showed mild to moderate pancolitis with a normal terminal ileum. He then presented to Ophthalmology with a 10-month history of bilateral blurry vision, erythema, irritation, photophobia, and diplopia. On exam, he was noted to have decreased visual acuity 20/80 bilaterally, left superior temporal and nasal field deficiencies, right optic disc pallor and left optic nerve swelling. A diagnosis of anterior uveitis was made and patient referred for evaluation by neuro-ophthalmology, who confirmed a diagnosis of optic disc neuritis, which was attributed to chronic TNF inhibitor therapy. A previous work up with a MRI of the brain and temporal artery biopsies had been non-diagnostic. He was diagnosed with anterior uveitis and was started on Prednisolone eye drops. The Adalimumab was discontinued, and the patient declined Vedolizumab as an alternative therapy. Most recently, he had one acute episode of vision loss in February of 2019 that was treated with IVIG but reports stabilized visual acuity and eye pain since then.

DISCUSSION:

There have been rare reports of approximately 1 in 1000 patients using TNF inhibitor drugs who experience de-novo or unmasking of latent demyelinating disease including optic neuritis. Both partial improvement and complete resolution of symptoms from optic neuritis has been reported upon discontinuation of TNF inhibitor therapy. Our patient continues to be off all biologics and his vision continues to improve.
2100 Linear IgA Bullous Dermatosis, Ulcerative Colitis, and Autoimmune Hepatitis: A Unique Triad
Authors: Kothari Shana , Berkelhammer Charles
Published: October 2019

INTRODUCTION:

Linear immunoglobulin IgA bullous dermatosis (LABD) is an autoimmune bullous disease characterized by formation of subepidermal blisters and linear deposition of IgA antibodies along the basement membrane zone. The link between LABD and ulcerative colitis (UC) has been previously documented and hypothesized to be due to immune exposure to autoantigens present in the colon, and subsequent targeting of these autoantigens in the skin. However, there have been no associated cases with autoimmune hepatitis. We report a unique triad of all three autoimmune conditions that presented concomitantly.

CASE DESCRIPTION/METHODS:

A 21-year-old male presented with vesicular and pustular rash on the skin of the hands, arms, abdomen, and legs (Figure 1). Histopathology revealed subepidermal blister with perivascular inflammation and linear C3 and IgA deposition along dermoepidermal junction consistent with LABD. The rash did not respond to topical corticosteroids. Liver enzymes were markedly elevated (AST 535 unit/L, ALT 436 unit/L, ALP 292 unit/L, bilirubin 0.3 mg/dL). He had iron deficiency anemia (Hgb 10.9 gm/dL, MCV 72.3 FL) with low ferritin. Autoimmune markers including antinuclear antibody, anti-smooth muscle, F-actin, anti-mitochondrial antibody, anti-liver kidney microsomal antibody, and anti-neutrophil cytoplasmic antibody were negative. Viral markers including Epstein Barr virus, cytomegalovirus, hepatitis A, B, C, E were negative, as were markers of metabolic liver disease. Celiac serologies were negative. Liver ultrasound was significant for hepatomegaly. Upper endoscopy with small bowel biopsies were normal. Colonoscopy revealed a patchy pancolitis consistent with ulcerative colitis, confirmed on biopsies. Prednisone 40 mg daily was administered, resulting in rapid resolution of the rash and elevated liver enzymes. Rapid improvement of transaminases with corticosteroids favors seronegative autoimmune hepatitis. He remains in clinical remission of his LABD, ulcerative colitis, and seronegative autoimmune hepatitis after gradual tapering off corticosteroids and on mesalamine alone.

DISCUSSION:

This is the first case, to our knowledge, where LABD, UC, and autoimmune hepatitis coexisted and had a successful therapeutic response to systemic corticosteroids. It is imperative to consider concomitant autoimmune diseases when a patient presents with such skin manifestations in order to guide early treatment.
2101 Non-Necrotizing Granulomas of the GI Tract and Abdominal Lymph Nodes. Crohn’s Disease, Sarcoidosis or Both?
Authors: Douglas Scott R. , Lotun Nitisha , Moore Danielle , Doran Amy , Hays R. Ann
Published: October 2019

INTRODUCTION:

Digestive tract involvement of sarcoidosis is rare (<2%) with few case reports of gastric and colon involvement and requires the presence of non-necrotizing granulomas. Non-necrotizing granulomas of the GI tract are typically associated with either Crohn’s disease or sarcoidosis. Distinguishing between the two can be a diagnostic dilemma.

CASE DESCRIPTION/METHODS:

A 25 y.o African American male presented with nausea, vomiting and weight loss. CT showed diffuse nodular airspace disease, lymphadenopathy, and thickening of the antrum and proximal duodenum. Lymphadenopathy involved the porta hepatis, stomach, head of the pancreas, abdominal mesentery, retroperitoneum and groin. EGD/EUS revealed mild erosive gastritis and duodenitis. Gastric and duodenal biopsies revealed non-necrotizing granulomas. Lymph node FNA showed reactive lymphocytes and non-necrotizing granulomas. Flow cytometry was normal. Colonoscopy with biopsy found nodular lymphoid hyperplasia in the terminal ileum (TI). Serum ACE level, lysozyme, and IgG4 were normal. IgG 3 was elevated. PFTs showed mildly reduced DLCO. Infectious work up was negative. He was treated with steroids and improved clinically. Three years later he presented with recurrent epigastric pain, anorexia, early satiety and profound weight loss. CT revealed increased abdominal lymphadenopathy. EGD showed inflammation and thickening of the stomach and duodenum. CSY revealed TI lymphoid hyperplasia, erosions over the IC valve and mild right sided colitis (Figure 1). Histology showed non-necrotizing granulomas with acute/chronic inflammation in the right colon, duodenum and stomach. CT chest showed regression of findings compared to 2016. Work-up revealed normal amyloid, ACE and soluble IL2 Ab receptor. Hypergammaglobulemia and hypoalbuminemia were present. He was treated with prednisone with rapid improvement.

DISCUSSION:

Our case demonstrates difficulty clarifying the diagnosis of non-necrotizing granulomas and distinguishing between atypical Crohn’s and sarcoidosis. Evidence of pulmonary involvement, low DLCO, lymphadenopathy and hypergammaglobulinemia argue in favor of sarcoidosis. Progression of inflammation to the TI and colon suggest Crohn’s. Pathology noted that granulomas were more abundant than commonly seen with Crohn’s disease alone. Additionally, there was more background inflammation than typically seen in GI sarcoid alone. A multidisciplinary team determined that this was consistent with Crohn’s and sarcoidosis occurring concomitantly.
2102 Crohn’s Disease With Rare Extra-Intestinal Manifestation and Unusual Great Outcome!!
Authors: Al Hillan Alsadiq , Hawa Fadi , Mohamed Mujtaba , Turtel Penny
Published: October 2019

INTRODUCTION:

Crohn’s disease (CD) is a chronic relapsing inflammatory disorder that most commonly affects the small and large bowel. Prolonged diarrhea with abdominal pain, with or without gross bleeding, fatigue, and weight loss are the hallmarks of Crohn’s disease. Extraintestinal manifestations, such as joints, eye and skin disorders, biliary tract involvement, and kidney stones, may occur. Tonsillar involvement in CD has been very rarely described in the literature. CD is diagnosed with endoscopic and imaging studies of the bowel in a patient with a compatible clinical history. We present a 29-year-old female patient who had a rare extra-intentional manifestation of Crohn's disease which is granulomatous tonsillitis and had an unexpectedly favorable outcome post-tonsillectomy being off treatment for 9 years now without any complaints.

CASE DESCRIPTION/METHODS:

A 29-year-old with no past medical history presented with frequent episodes of tonsillitis followed by prolonged diarrhea with abdominal pain. Physical exam showed enlarged tonsils and generalized lower abdominal tenderness. Labs showed elevated C-reactive protein, Sedimentation rate, and fecal calprotectin. The patient underwent colonoscopy that showed scattered ileocolitis and biopsy results were compatible with Crohn’s disease. Patient’s tonsillitis failed multiple courses of antibiotic therapy. Accordingly, tonsillectomy was performed, and biopsy showed granulomatous lesion consistent with Crohn’s disease. The patient has been off treatment and never required maintenance therapy for 9 years now without any complaints.

DISCUSSION:

The first case of CD with granulomatous tonsillitis was reported in 1984. CD patients with an oropharyngeal manifestation of their disease are rare, with an estimation of only 0.5%–20% of patients, with a higher incidence in younger patients. A systematic review and meta-analysis did by Sun et al. demonstrated that tonsillectomy in healthy individuals is associated with an increased risk of developing Crohn’s Disease. Koutroubakis et al. did a case-control study that also showed the same results. Our case presented with recurrent tonsillar pain initially which is a rare extra-intestinal manifestation and underwent tonsillectomy with biopsy confirming Crohn’s Disease and had a remarkably unusual recovery from all Crohn’s Disease symptoms and never had any complaints in 9 years off medical treatment.