Multiple Intestinal Ulcers Research Articles

P358 Long-term clinical outcomes of intestinal Behçet’s disease: a 30-year cohort study at a tertiary hospital in South Korea

Abstract Background Long-term clinical data on intestinal Behçet’s disease (BD) are scarce because of the rarity of this disease, and many issues remain unresolved due to the difficulty in performing a well-designed clinical trial or epidemiologic study. We aimed to investigate the long-term clinical outcomes of patients with intestinal BD with a 30-year cohort study at a tertiary hospital in South Korea, a hospital with the largest number of patients with intestinal BD worldwide. Additionally, we aimed to identify factors that could predict various clinical outcomes, including anti-tumor necrosis factor-alpha (anti-TNF-α) agent use, intestinal surgery, hospitalization, and emergency room visits. Methods A cohort of 780 patients with intestinal BD between 1997 and 2021 was investigated to determine the long-term clinical outcomes and prognostic factors at the inflammatory bowel disease Clinic of Severance Hospital, Seoul, Korea. Results During the median follow-up period of 12.7 ± 7.2 years, 5-aminosalicylic acids, corticosteroids, immunomodulators, and anti-tumor necrosis factor-alpha (TNF-α) agents were required in 94.9%, 67.2%, 43.8%, and 14.6% of the patients, respectively. The cumulative rates of anti-TNF-α use were 3.7%, 7.5%, 8.5%, 12.1%, 17.6%, and 24.0%, and that of abdominal surgery were 5.7%, 10.9%, 12.6%, 16.5%, 21.6%, and 28.3%, at 1, 3, 5, 10, 20, and 30 years, respectively, after the initial diagnosis of intestinal BD. The cumulative rates of hospitalization were 11.8%, 21.9%, 27.9%, 38.8%, 54.4%, and 74.8% and that of emergency room visits were 10.0%, 19.8%, 22.7%, 31.6%, 50.0%, and 65.0% at 1, 3, 5, 10, 20, 30 years. Older age at primary diagnosis, previous appendectomy history, higher disease activity index for intestinal Behçet’s disease score, systemic BD, multiple intestinal ulcers, deep intestinal ulcers, higher C-reactive protein, lower hemoglobin, and lower albumin levels were associated with poor prognosis. Married status, higher body mass index, oral ulceration, and arthritis were negatively associated with a poor prognosis. Conclusion The data on the long-term clinical outcomes of intestinal BD and their prognostic factors could guide physicians for adequate patient monitoring and optimizing individualized treatments.

Long-term clinical outcomes of intestinal Behçet's disease: A 30-year cohort study at a tertiary hospital in South Korea.

We aimed to identify the long-term clinical outcomes of and prognostic factors for intestinal Behçet's disease (BD). A cohort of 780 patients with intestinal BD between 1997 and 2021 was investigated to determine long-term clinical outcomes and prognostic factors at an inflammatory bowel disease clinic at Severance Hospital, Seoul, Korea. During the median follow-up period of 12.7±7.2years, 5-aminosalicylic acids, corticosteroids, immunomodulators, and anti-tumor necrosis factor-alpha (TNF-α) agents were required in 94.9%, 67.2%, 43.8%, and 14.6% of the patients, respectively. The cumulative rates of anti-TNF-α use were 3.7%, 7.5%, 8.5%, 12.1%, 17.6%, and 24.0%, and those for abdominal surgery were 5.7%, 10.9%, 12.6%, 16.5%, 21.6%, and 28.3%, at 1, 3, 5, 10, 20, and 30years, respectively, after initial diagnosis of intestinal BD. The cumulative rates of hospitalization were 11.8%, 21.9%, 27.9%, 38.8%, 54.4%, and 74.8%, and those of emergency room visits were 10.0%, 19.8%, 22.7%, 31.6%, 50.0%, and 65.0% at 1, 3, 5, 10, 20, and 30years. Older age at primary diagnosis, previous appendectomy history, higher disease activity index for intestinal Behçet's disease score, systemic BD, multiple intestinal ulcers, deep intestinal ulcers, higher C-reactive protein, lower hemoglobin, and lower albumin levels were associated with poor prognosis. Married status, higher body mass index, oral ulceration, and arthritis were negatively associated with poor prognosis. Data on the long-term clinical outcomes of intestinal BD and their prognostic factors could guide physicians in patient monitoring and in optimizing individualized treatment.

S1969 Treatment of Refractory ICI-Associated Colitis With Fecal Microbiota Transplant

Introduction: The clinical benefits of immune checkpoint inhibitors (ICIs) have been associated with immune-related adverse events (irAEs), with ICI-associated colitis among the most reported. ICI-associated colitis is routinely treated with immunosuppressive therapy, including corticosteroids and anti-TNF agents. Recent studies have suggested fecal microbiota transplant as a potential treatment for refractory ICI-associated colitis. Case Description/Methods: Middle aged female with multiple sclerosis in remission and stage IV melanoma with bone and pulmonary involvement who was first diagnosed about 3 years ago. She underwent palliative radiation followed by 4 cycles of ipilimumab and nivolumab initially. Despite this therapy she had cancer progression. She was then started on treatment with cisplatin, vinblastine and dacarbazine (CVD), however she failed to respond. She was switched to targeted therapy with encorafenib and binimitinib. Within 2 months, she was admitted to the hospital with anemia and hypotension, endoscopic evaluation revealed multiple intestinal and colonic ulcers. Based on these findings she was suspected to have ICI enterocolitis and received 2 cycles of infliximab plus high dose corticosteroids. She resumed encorafenib and binimitinib after clinical improvement and started single agent nivolumab. She was readmitted within 3 months and again at 4 months after for anemia and melena despite receiving additional treatment with vedolizumab and multiple courses of high dose corticosteroids. She was then treated with ustekinumab; however, she was again hospitalized a fourth time for similar symptoms. Decision was made to treat her refractory colitis with fecal microbiota transplant (FMT) as compassionate use. She had a remarkable response, with complete resolution of her GI symptoms with no additional immunosuppressive therapy or hospitalizations. However, patient’s cancer continued to progress. Her course was further complicated by oculomotor nerve dysfunction and ultimately was transitioned to hospice care and passed away 4 months later (Figure). Discussion: Fecal microbiota transplant has emerged as novel therapeutic option for treatment of refractory ICI-associated colitis. Combination treatment with ICI and targeted therapy may be a risk factor for more severe colitis with significant ulcers and refractory disease course. Further studies are needed to assess the risk factors related to combination therapy and guide more effective treatment.Figure 1.: A: Altered anatomy near cecum, B: Ulcer in terminal ileum, C: Ulcerated mass in ascending colon, D: Ascending colon narrowing.

Deficiency of adenosine deaminase 2 in an elderly female with multiple intestinal ulcers and hemorrhagic stroke

Deficiency of adenosine deaminase 2 in an elderly female with multiple intestinal ulcers and hemorrhagic stroke

Assessment of Talaromyces Marneffei Infection of the Intestine in Three Patients and a Systematic Review of Case Reports.

BackgroundHematogenous dissemination of Talaromyces marneffei can result in multiorgan involvement (skin, lung, and reticuloendothelial system involvement); however, few studies have reported intestinal T marneffei infections. We investigated clinical features, management, and patient outcomes concerning Talaromyces-related intestinal infections.MethodsPatients with Talaromycosis between August 2012 and April 2019 at The First Affiliated Hospital of Guangxi Medical University, China, were retrospectively analyzed. Patients presenting with intestinal Talaromycosis and endoscopy-confirmed diagnoses were investigated. We also undertook a systematic review of the relevant English and Chinese literature.ResultsOf 175 patients diagnosed with Talaromycosis, 33 presented with gastrointestinal symptoms, and 31 underwent stool cultures, 1 of which tested positive. Three patients had gastrointestinal symptoms and negative stool cultures, and endoscopic tissue biopsy confirmed a pathological diagnosis. A systematic review of 14 reports on human Talaromycosis identified an additional 16 patients. Fever, weight loss, and anemia were the most common symptoms, along with abdominal pain, diarrhea, and bloody stools. Abdominal computed tomography showed intestinal wall edema and thickening and/or abdominal lymphadenopathy. Endoscopy showed erosion, hyperemia, edema, and multiple intestinal mucosal ulcers. Of the 19 patients, 16 received antifungal therapy, 14 of whom recovered and 2 died. Three patients received no therapy and died.ConclusionsGastrointestinal disseminated Talaromycosis is not rare and can affect the stomach, duodenum, and colon, and may involve the entire digestive tract. Colon is the most common site. Endoscopy is needed for patients presenting with gastrointestinal symptoms in T marneffei-infected endemic areas. Systemic application of effective antifungal therapy can improve the prognosis.

Multiple Intestinal Ulcers Associated with Primary Epstein-Barr Virus Infection in a Patient with Rheumatoid Arthritis Undergoing Methotrexate Therapy.

A 47-year-old woman with a 2-year history of rheumatoid arthritis (RA) undergoing methotrexate treatment developed a perforated ulcer in the ileum for which she underwent emergency surgery. A histological analysis of the extirpated specimen presented a possible Epstein-Barr virus (EBV) infection in the ulcerative lesion without a feature of lymphoproliferative disorder. Interestingly, the patient's serological tests with a paired serum diagnosed a primary EBV infection. The present case emphasizes the importance of being aware of severe enteritis as a possibility for patients with RA, for an accurate diagnosis.

Ulcérations buccales aphtoïdes persistantes associées au tocilizumab : deux cas

Tocilizumab, a humanized monoclonal antibody that blocks interleukin-6 receptor, is approved for use in rheumatological diseases. The most frequent adverse events reported are infections. We describe for the first time the occurrence of recurrent aphthous mouth ulcers in two patients on TCZ. Two patients were treated with TCZ for rheumatological disease. A few weeks after administration of TCZ, they presented with multiple painful mouth ulcers that would not heal until TCZ had been withdrawn. In both cases, the oral ulcers resolved 6 to 7weeks after withdrawal of TCZ and readministration of the drug led to recurrence of oral ulcers within 10days in both patients. We describe for the first time the occurrence of aphthous mouth ulcers induced by TCZ. The causative role of TCZ was established by positive rechallenge. These cases were similar to a recently reported case of multiple intestinal aphthous ulcers occurring during TCZ treatment. Such mucosal side effects may be explained by similar inflammatory mechanisms but appear paradoxical because TCZ inhibits a pro-inflammatory cytokine, interleukin 6. TCZ treatment can be maintained if necessary, in combination with colchicine, as reported for one of our patients.

An Uncommon Cause of Severe Epigastric Pain with Multiple Intestinal Ulcers after Renal Transplantation- Strongyloides Hyperinfection

An Uncommon Cause of Severe Epigastric Pain with Multiple Intestinal Ulcers after Renal Transplantation- Strongyloides Hyperinfection

EBV-associated lymphoproliferative disorders misdiagnosed as Crohn's disease

Epstein–Barr virus (EBV) plays an etiological role in various diseases. EBV-associated lymphoproliferative disorder (LPD) is usually observed in individuals with congenital or acquired immune deficiencies but was also recently reported in non-immunocompromised individuals. Two cases of immunocompetent patients with EBV-associated T-cell LPD of the small bowel and colon who were initially misdiagnosed as Crohn's disease (CD) are reported here. EBV-associated T-cell LPD with primary gastrointestinal tract involvement can manifest as multiple discrete ulcers of the small and/or large bowel that are similar to the lesions found in CD or intestinal tuberculosis. However, when patients have multiple intestinal ulcers that are not typical of CD or intestinal tuberculosis and the clinical course is unusual, clinicians should consider the possibility of EBV-associated LPD that involves the gastrointestinal tract because the treatment strategy and prognosis are completely different.

Toxic epidermal necrolysis complicated by multiple intestinal ulcers

On 11 November 2001, a 30-year-old woman was treated by an emergency doctor for cold symptoms, including pyrexia and pain in the pharynx. She was prescribed norfloxacin (NFLX), acetaminophen, and tranexamic acid, as well as additional supporting medicines that she chose not to use. On the following day, the patient went to the outpatient unit of the department of internal medicine of a general hospital; she was admitted because of continuing fever. She was prescribed a combination medication for Bifidus bacillus containing mefenamic acid and aluminum hydroxide gel/magnesium hydroxide, as well as NFLX and teprenone, but erosions of her face appeared on 15 November, followed by erosions of the trunk on 17 November. She was suffering from inflammation and blisters over her whole body. She was diagnosed with toxic epidermal necrolysis (TEN) by the department of dermatology of the same hospital. The patient underwent steroid pulse therapy for 3 days from 17 November, which was not effective, and was transferred to our hospital on 22 November. In the initial examination, the patient's body was almost entirely covered with blisters, including her face, trunk, and both arms. Erosions were also present in her mouth, on the conjunctivae of both eyelids, and on her vulva (see Fig. 1). In addition, we recognized false membrane formation on the eyelid conjunctivae in both corneas. Abrasions covered 61% of the patient's body. Figure 1Open in figure viewerPowerPoint The patient's body was almost entirely covered with blisters, including her face (a), trunk (b), and both arms (c). Erosions were also present in her mouth, on the conjunctivae of both eyelids, and on her vulva The patient's white blood cells were in the normal range, but impaired liver function and hyperbilirubinemia were recognized, and tests for various virus antibodies showed no meaningful increase. A skin biopsy was performed on the dot-shaped erythema exudativum present on the patient's lower limb. This specimen revealed the formation of cuticle lower part blister eosinophil necrosis, and slight lymphocyte inflammation of blood vessel circumference of upper dermis (Fig. 2). Figure 2Open in figure viewerPowerPoint Skin biopsy specimen from the dot-shaped erythema exsudativum present on the patient's right lower limb (hematoxylin and eosin stain) The patient was diagnosed with TEN from the clinical features of acute and widespread epidermal sloughing, the clinical history, and pathologic findings of necrosis and dermoepidermal separation. She was treated three times with plasma exchange on 22, 23, and 26 November. In addition, the procedures recommended for second-degree scalding burns were executed, using a burn bed and providing daily external treatment with a povidone iodine and polymyxin B sulfate ointment. Because the patient was not able to eat orally, intravenous hyperalimentation (IVH) management was conducted and antibiotics (fosfomycin, imipenem cilastatin, vancomycin) were administered for secondary infection prevention. The false membrane of the eye was removed. Epidermis formation resumed, and the patient gradually recovered, including recovery of her eyesight on the 14th disease day. Despite these recoveries, the patient's diarrhea persisted and there was blood in her stool. Therefore, intestinal and colon endoscopies were performed (Fig. 3). The endoscope was inserted to about 40 cm in the upper part of the jejunum, determined by the disappearance of the kerckring fold. A circular ulcer and general detachment of the villus were identified, as well as an edematous structure on the mucous membrane of the intestine. An examination of the colon revealed no active ulcers, but there was evidence of a healing ulcer. The patient was diagnosed with multiple small intestinal ulcers complicated by TEN. IVH management was performed again to treat the intestinal ulcers. The patient's diarrhea decreased slowly, and it had decreased to a single occurrence per day in early February 2002. Figure 3Open in figure viewerPowerPoint The endoscopic image showed multiple ulcers of the intestine. A circular ulcer, the general detachment of the villus, the disappearance of the kerckring fold, and an edematous structure on the mucous membrane of the intestine were revealed Fluid food was initiated on 4 February. Ileus was recognized shortly thereafter and ingestion was stopped temporarily on 9–13 February, and then reintroduced gradually. Improvement of the ulcer was confirmed by endoscopic findings on 8 March and, by the end of March, the patient was able to take foods orally. In addition to the above conditions, pleurisy of the left side was found in December. Thoracentesis and bronchoscopy were performed, but no cause was found. Because the patient's symptoms were slight, it was decided to keep her under observation for pleurisy. The patient left the hospital on 18 April. She no longer needed medication at that time. She was investigated by lymphocyte stimulation and by scratch-patch test; however, all the medicines used were negative by these tests.

A case of enterocutaneous fistula due to multiple intestinal ulcers in the blind loop

A case of enterocutaneous fistula due to multiple intestinal ulcers in the blind loop

Trisomy 8 Involved in Myelodysplastic Syndromes as a Risk Factor for Intestinal Ulcers and Thrombosis — Behçet's Syndrome

Only 12 myelodysplastic syndrome (MDS) cases with Behçet's syndrome have been previously reported and trisomy 8 was found to have accumulated in all these patients. Five of the cases had complications in the form of multiple intestinal ulcers, which is one of the symptoms of Behçet's syndrome. To investigate the relationship between trisomy 8 and multiple intestinal ulcers in MDS patients, we analyzed 46 MDS cases treated in our hospital over the last decade, and trisomy 8 was observed in eight of them. Three of these cases had complications of both multiple intestinal ulcers and thrombosis, and two cases showed episodes of thrombosis without intestinal ulcers. All these five cases featured trisomy 8, while the other 38 MDS patients without trisomy 8 had no episode of either intestinal ulcer or thrombosis. Two of the three cases suffering from multiple intestinal ulcers were treated with granulocyte-colony stimulating factor (G-CSF), which resulted in aggravation of the symptoms. Although the influence of G-CSF on such symptoms in MDS patients with trisomy 8 remains unclear, it seems advisable to exercise caution in the use of G-CSF when an MDS patient with trisomy 8 has intestinal ulcers or thrombosis.

A CASE OF HEMORRHAGIC MULTIPLE INTESTINAL ULCER IN THE BLIND LOOP

This paper describes a case of blind loop syndrome with repeated anal bleeding caused by multiple ulcer at the blind loop which was formed after a side to side anastomosis of the ileum was performed for intestinal obstruction 30 years before. A 62-year-old female who had undergone a bypass surgery for small bowel obstruction at the age of 30 was admitted to the hospital because of melana and lower abdominal pain. Small intestinal studies with barium, RI and angiography suggested bleeding from the ileum. Then the blind portion was resected and an end to end anastomesis was performed. The surgical specimen showed side to side anastomosis about 55cm from the terminal ileum, and a dilated blind loop. Multiple intestinal ulcers were seen in the blind loop which were histologically diagnosed as nonspecific ulcers. We have to entertain a possibility of bleeding from the blind loop, when the bleeding case of unknown origin following an intestinal surgery is encountered.

小腸穿孔と血球貧食性組織球増多をみたくすぶり型成人型T細胞性白血病リンパ腫の一剖検例

An autopsy case with atypical adult T cell leukemia lymphoma (ATL) was reported. A 24-year-old male who had skin lesions and respiratory distress as premonitory symptoms from his childhood. Skin lesions appeared in the form of vasculitis. Infiltration of ATL cells were not recognizable in the skin and peripheral blood. On his admission, before four months of his death, ATLA (antibody to ATL virus associated antigen) was detected to be positive in his serum and simultenously also in the serum of his mother whose native place south-west region of Japan (Kagawa prefecture). After admission, acute abdominal symptoms occurred and roentgenography revealed multiple intestinal ulcers. He was treated with surgical operations three times, however, massive alimentary bleeding has continued and died on July, 1, 1987.The infiltration of atypical lymphoid cell of T cell character (Leu 1 positive) was observed in the ulcerated walls of intestine surgically resected. Autopsy revealed systemic swelling of lymph nodes of their cell components being in agreement with picture of pleomorphic type of ATL Invasion of ATL cells were found in various organs, i.e. liver, kidneys, spleen, gastrointestinal tract, adrenals and bone marrow. Moreover, remarkable haemophagocytic cells appeared in the lymph nodes, spleen and liver. Lymphoid cells had pleomorphic contour and large, irregular shaped, convoluted nuclei. Immunocytochemically, these cells were positive for Leu 1 and faintly positive for OKIa. On the other hand, reaction for MT-1, MB-1, immunoglobulins and histiocytic markers were negative. Phagocytic histiocytes positively reacted for lysozyme, α1-antitrypsin, and CEA, but negatively for S100 protein. No pulmonary infection was recognized. From clinical and anatomical evidences the patient developed typical ATL having duration of more than ten years with insidious course but without leukemic manifestation. This picture corresponded to smoldering ATL proposed by Takatsuki and Yamaguchi.

Prevention of indomethacin-induced intestinal ulcers by spironolactone and norbolethone

Both spironolactone (an antimineralocorticoid compound) and norbolethone (an anabolic steroid) protect the rat against the production of multiple intestinal ulcers and peritonitis by indomethacin.