Salvage camrelizumab for an intractable NK/T cell lymphoma patient with two instances of intestinal perforation: a case report and literature review.

BACKGROUND: The prognosis of natural killer/T cell lymphoma (NKTCL) with multifocal small intestine involvement complicated by intestinal perforation is extremely poor. There is no evidence-based treatment strategy for this intractable condition. CASE PRESENTATION: A 30-year-old male was admitted to our hospital in April 2017 and presented with recurrent fever for three months and multiple painless subcutaneous nodules in the abdominal wall. An excision biopsy of the subcutaneous nodules in the abdominal wall revealed NKTCL. The patient was diagnosed with stage IVB NKTCL with skin and multifocal small intestinal involvement according to the imaging results. The first intestinal perforation occurred due to tumor infiltration before the initial treatment. The second intestinal perforation occurred after receiving two cycles of chemotherapy with a modified SMILE regimen. The histone deacetylase inhibitor (HDACi) chidamide was administered as a single-agent therapy after recovery from the second intestinal perforation. Complete remission was achieved. Unfortunately, five months later, the patient was confirmed to have relapsed and received the salvage chemotherapy. The patient suffered from disease progression again after the fourth cycle of chemotherapy. At this point, from May 29, 2018, the patient started to receive injections of the anti-programmed death 1 (PD-1) antibody camrelizumab as a salvage treatment. Two months after the initial anti-PD-1 antibody camrelizumab injection, the response was partial remission. Disease progression was confirmed in March 2021, with a progression-free survival time of 34 months. CONCLUSIONS: NKTCL patients with multifocal small intestine involvement have a high risk of intestinal perforation. The possible etiologies of bowel perforation include tumor infiltration, tumor necrosis in response to therapy, and acute inflammation. The anti-PD-1 antibody camrelizumab may be a new candidate agent for treating this type of intractable NKTCL. Further observations are necessary to identify the efficacy and safety of new agents in the future.

Characteristics, treatment, and outcome of patients with bowel perforation after immune checkpoint inhibitor exposure.

PURPOSE: Exposure to immune checkpoint inhibitors (ICIs) can predispose to immune-related adverse events (irAEs) involving the gastrointestinal tract. The association between ICIs and bowel perforation has not been well studied. We aimed to describe the clinical course, complications, treatment, and outcomes of patients experiencing bowel perforation during or after ICI treatment. METHODS: This retrospective, single-center study included adult cancer patients with bowel perforation that occurred between the first dose of ICI treatment and up to 1 year thereafter between 1/1/2010 and 4/30/2021. Patients' clinical course, imaging, treatment, and outcomes related to bowel perforation were collected and analyzed. RESULTS: Of the 13,991 patients who received ICIs during the study period, 90 (0.6%) met the inclusion criteria. A majority were male (54.4%), the most common cancer type was melanoma (23.3%), and most patients had received PD-1/L1 inhibitor treatment (58.8%). Onset of perforation occurred after a median of four ICI treatment cycles. The most common symptom was abdominal pain (95.5%). The colon was the most common location for the perforation (37.7%). Evidence of diverticulitis, enterocolitis, or appendicitis was seen in 32 (35.6%) patients, and 6 (6.6%) patients had luminal cancer involvement at the time of perforation. The overall hospitalization rate related to perforation was 95.5%, with mortality of 15.5% during the same admission. Antibiotics were given in 95% of our sample; 37.8% of patients also required surgical/interventional radiology intervention. Forty-six patients (51.1%) had perforation-related complications (e.g., sepsis, fistula, abscess), which were associated with a higher mortality rate (30%). CONCLUSION: Our findings suggest a low incidence of bowel perforation after ICI treatment (0.6%), with 40% of patients having coexisting bowel inflammation as a potential contributing factor. Patients with bowel perforation had an aggressive disease course and high rates of hospitalization, complications, and mortality. Early recognition and prompt intervention is critical to improve patient outcomes. Future studies are warranted to further investigate the cause, predictive markers, and optimal treatment for this patient population.

Intestinal perforation after intravitreal low dose ranibizumab injection for the treatment of type 1 retinopathy of prematurity: A case report.

PURPOSE: To report a newborn patient with gastrointestinal (GIS) perforation after intravitreal ranibizumab (RBZ) treatment. CASE REPORT: The patient was born at 31 gestational week and hospitalized with the diagnosis of small for gestational age and prematurity. In the follow up he underwent GIS surgery due to necrotizing enterocolitis (NEC) and was diagnosed with retinopathy of prematurity (ROP). At 43 weeks of postmenstrual age, he developed intestinal perforation after 12 h of the second low-dose RBZ injection. According to our knowledge, this is the first report of GIS perforation due to low-dose intravitreal RBZ treatment in an infant with severe ROP. CONCLUSION: The risk of GIS perforation should be taken into consideration during the application of intravitreal vascular endothelial growth factor antagonist agents, especially in newborns with previous GIS surgery and a history of NEC, and these patients should be carefully monitored for GIS complications.

Bowel obstruction and perforation during pregnancy: Case report and literature review.

OBJECTIVE: Bowel perforation during pregnancy is a rare but life-threatening situation, and the management remained uncertain. We presented a case in our hospital and reviewed literatures to provide a guidance to deal with this challenging situation. CASE REPORT: A case at gestational age 26 weeks and 4 days, who had bowel perforation over previous ileo-colonic anastomosis site. The fetus was delivered via cesarean section after 1 week of operation and both were discharged without major complication at 35 days after delivery. We searched Pubmed and reviewed 15 English literatures and make a discussion. CONCLUSION: A simultaneous cesarean section could be considered if the gestational age was large enough to avoid the comorbidity of preterm birth or the maternal risk of sepsis outweigh the risk of preterm birth. Otherwise, tocolysis should be used in an attempt to delay delivery until the antenatal corticosteroids have worked.

Bowel perforation due to methotrexate therapeutic error: an insidious adverse reaction.

OBJECTIVE: Methotrexate (MTX) is widely used in the treatment of rheumatic and non-rheumatic disorders. Severe adverse effects are often associated with therapeutic errors, such as daily intake rather than weekly intake. Among them, the risk of bowel perforation is extremely rare (0.1%). We describe a case of bowel perforation, occurred following daily intake of MTX. CASE REPORT: A 68-year-old man was prescribed to take MTX 7,5 mg orally once a week, while waiting for switch to abatacept for a recent reactivation of rheumatoid arthritis. After 10 days he started having pharyngodynia, hematochezia and general malaise. At medical examination he presented oral and nasal mucositis; moreover, blood exams showed thrombocytopenia. The anamnesis revealed that he had been taken the prescribed dosage of MTX daily, instead of weekly. Therapy with Lederfolin 1000 mg (mg/m²/die) and urine alkalinization started. After 7 days of hospitalization, there was an abrupt worsening of clinical conditions and an emergency CT scan revealed millimetric gas bubbles indicating bowel perforation. The patient underwent an emergency exploratory laparotomy that resulted in peritoneal toilette and sigma resections. Anatomopathological findings were suggestive of MTX poisoning. CONCLUSIONS: The patient was discharged on the 17th day in good clinical condition.

Intravenous versus intravenous/oral antibiotics for perforated appendicitis in pediatric patients: a systematic review and meta-analysis.

BACKGROUND: The use of oral (PO) antibiotics following a course of certain intravenous (IV) antibiotics is proposed in order to avoid the complications of IV medications and to decrease the cost. However, the efficacy and safety of sequential IV/PO antibiotics is unclear and requires further study. METHODS: The databases, including PubMed, EMBASE and Cochrane Library, were searched. Studies comparing outcomes in patients with perforated appendicitis receiving sequential IV/PO and PO antibiotics therapy were screened. The Newcastle-Ottawa Scale (NOS) and the Jadad score were used to evaluate the quality of the cohort and the randomized controlled portions of the trial, respectively. Statistical heterogeneity was assessed using the I2 value. A fixed or random-effect model was applied according to the I2 value. RESULTS: Five controlled studies including a total of 580 patients were evaluated. The pooled estimates revealed that sequential IV/PO antibiotic therapy did not increase the risk of complications, with a risk ratio (RR) of 0.97 (95% CI 0.51-1.83, P = 0.93) for postoperative abscess, 1.04 (95% CI 0.25-4.36, P = 0.96) for wound infection and 0.62 (95% CI 0.33-1.16, P = 0.13) for readmission. CONCLUSIONS: Our study demonstrates that sequential IV/PO antibiotic therapy is noninferior to IV antibiotic therapy regarding postoperative abscess, wound infection and readmission.

Small bowel perforation secondary to CMV-positive terminal ileitis postrenal transplant.

Cytomegalovirus (CMV) infection of the gastrointestinal tract is common in immunosuppressed patients; however, small bowel perforation from tissue-invasive CMV disease after many years of immunosuppressive therapy is a rare complication requiring timely medical and surgical intervention. We report a case of a postrenal transplant patient who presented to the emergency department with severe lower abdominal pain with CT of the abdomen/pelvis revealing a small bowel perforation. He underwent an emergent laparoscopic right hemicolectomy, and his histopathology of the terminal ileum was positive for CMV disease. He was successfully treated with intravenous ganciclovir postoperatively. We discuss the pathophysiology, histopathological features and treatment of CMV infection.

Fístula duodeno-cava tras tratamiento con bevacizumab / Duodenocaval fistula after treatment with bevacizumab

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Synergistic effect of enteral nutrition on remission induction in a patient with penetrating Crohn disease: A case report.

RATIONALE: Crohn disease includes 3 phenotypes, inflammatory, stricturing, and penetrating. In cases where corticosteroids and immunosuppressive agents are not suitable treatment options, enteral nutrition (EN) can be used to reduce disease severity and enhance barrier defense with fewer potential adverse effects. PATIENT CONCERNS: A 23-year-old man with abdominal pain and diarrhea presented at our hospital in 2014. The frequency of defecation was 3 or 4 times a day without mucus or blood in the stool. His body mass index was 15.8, and in laboratory tests the erythrocyte sedimentation rate was 42.4 mm/h, serum C reactive protein was 65.2 mg/L, the leukocyte count was 11.64 × 109/L, and hemoglobin was 111 g/L. DIAGNOSIS: In computed tomography (CT) enterography the ascending colon was thickened, and there was effusion and enlarged lymph nodes around the colon. Colonoscopy revealed ulcer, polypoid proliferation, and bowel stenosis in many segments. Chronic inflammation was evident in multiple biopsies. Crohn disease was diagnosed based on the above observations. INTERVENTIONS: Mesalazine was administered at a dose of 4 g daily for 2 years. The patient was hospitalized again due to severe abdominal pain and ongoing fever. Intestinal perforation was detected via CT. Percutaneous drainage was performed followed by administration of intravenous metronidazole (0.5 g) and ciprofloxacin (0.2 g) twice a day. Peptison liquid was used as exclusive EN. After 2 weeks the antibiotics regimen was changed to metronidazole 0.4 g twice a day and ciprofloxacin 0.25 g 3 times a day, both administered orally. OUTCOMES: CT revealed that the infection was eliminated and the fistula was healed after 10 weeks, at which point antibiotics and exclusive EN was discontinued. Azathioprine was prescribed at a dose of 2 mg/kg daily to maintain clinical remission. The patient did not report any pain or diarrhea at a 1-year follow-up visit. LESSONS: The present case suggests that exclusive EN combined with antibiotics is useful in inducing remission in Crohn disease patients with active disease and penetrating complications.

Non-operatively managed case of contained jejunal diverticular perforation.

Jejunal diverticulosis is an underdiagnosed condition due to its relatively benign existence and uncharacteristic presentation. The complications can be very severe and, due to its often late diagnosis, patients may require urgent surgery. We present a woman who initially complained of non-specific abdominal symptoms but was diagnosed with a contained jejunal diverticular perforation relatively early. We managed her non-operatively with intravenous antibiotics from which she recovered well. She was discharged 2 days later and has remained completely well. Follow-up at 3 months showed no recurrence. Our case differs from most of the literature due to the early diagnosis and successful non-operative management of the patient. We conclude that, in cases of non-specific abdominal pain with diagnostic ambiguity, a diagnosis of small bowel diverticulosis should be considered. It should be managed non-operatively where possible.

Intestinal Behçet's Disease with Primary Myelofibrosis Involving Trisomy 8.

Behçet's disease (BD) is a disorder characterized by systemic inflammation of multiple organs, including the intestines. Several studies have reported a relationship between myelodysplastic syndrome and BD, and trisomy 8 was frequently seen, especially in intestinal BD. However, the association of BD with primary myelofibrosis (PMF) has not been well documented. A 58-year-old Japanese female was diagnosed with PMF in 2014. The symptoms of PMF resolved with ruxolitinib. However, she developed fever and intestinal perforation due to multiple ulcers in the terminal ileum in 2017. Intestinal perforation recurred 1 month later, and the dose of ruxolitinib was tapered. After discontinuation of ruxolitinib, she presented with recurrent oral aphthous ulcers and uveitis. Subsequently, intestinal perforation recurred, and she was diagnosed with intestinal BD. Trisomy 8 was identified in her peripheral blood. She underwent steroid therapy, azathioprine, and infliximab. This case suggests relationships between PMF, trisomy 8, and BD.

Not your usual hip pain: necrotising fasciitis secondary to sigmoid perforation.

Necrotising fasciitis (NF) resulting from sigmoid perforation is rare, but none the less remains life threatening. Early surgical intervention and empiric broad-spectrum antibiotic remains the standard of care. A 65-year-old man with history of stage 4 rectal cancer presented with bilateral hip pain. An X-ray of the hip and pelvis to rule out fracture revealed abnormal gas pattern overlying the right hip. CT imaging revealed distal sigmoid perforation with air extending from the pelvis to posterior aspect of the right hip and thigh. Aggressive surgical intervention and antibiotic initiation was associated with successful outcomes in our patient. Although less common, sigmoid perforation leading to NF in posterior thigh is a reminder of a potentially lethal complication.

European Paediatric Surgeons' Association Survey on the Management of Pediatric Appendicitis.

AIM: To define patterns in the management of pediatric appendicitis. METHODS: A total of 169 delegates from 42 (24 European) countries completed a validated survey administered at the EUPSA 2017 annual congress. RESULTS: In the work-up of children with suspected acute appendicitis, most surgeons rely on full blood count (92%), C-reactive protein (82%), and abdominal ultrasonography (76%), but rarely on computed tomography scans or magnetic resonance imaging. In suspected simple appendicitis, most surgeons (76%) do not perform appendectomy at night in clinically stable patients and start antibiotic preoperatively (64%), but only 15% offer antibiotic therapy alone (no appendectomy). In suspected perforated appendicitis, 96% start antibiotic preoperatively, and 92% perform an appendectomy. Presence of phlegmon/abscess is the main contraindication to immediate surgery. In case of appendix mass, most responders (75%) favor a conservative approach and perform interval appendectomy always (56%) or in selected cases (38%) between 2 and 6 months from the first episode (81%). Children with large intraperitoneal abscesses are managed by percutaneous drainage (59% responders) and by surgery (37% responders). Laparoscopy is the preferred surgical approach for both simple (89%) and perforated appendicitis (81%). Most surgeons send the appendix for histology (96%) and pus for microbiology, if present (78%). At the end of the operation, 58% irrigate the abdominal cavity only if contaminated using saline solution (93%). In selected cases, 52% leave a drain in situ. CONCLUSION: Some aspects of appendicitis management lack consensus, particularly appendix mass and intraperitoneal abscess. Evidence-based guidelines should be developed, which may help standardize care and improve clinical outcomes.

Caffeine exposure and acute kidney injury in premature infants with necrotizing enterocolitis and spontaneous intestinal perforation.

BACKGROUND: Acute kidney injury (AKI) is common in preterm infants, but specific therapies remain scarce. Recent studies have demonstrated an association between caffeine exposure and less frequent AKI in the first 7-10 days after birth. We hypothesized that patients with necrotizing enterocolitis (NEC) and spontaneous intestinal perforation (SIP) would provide a better natural model of AKI to evaluate this association. METHODS: We reviewed all premature patients diagnosed with NEC or SIP at our institution from 2008 to 2014. AKI was defined by change in serum creatinine using the neonatal Kidney Disease: Improving Global Outcomes definition. Caffeine was prescribed for apnea of prematurity and caffeine exposure was determined by chart review. RESULTS: A total of 146 patients with NEC/SIP were reviewed. Of these, 119 (81.5%) received caffeine, and 91 (62.3%) developed AKI. AKI occurred less frequently in patients who received caffeine than in those who did not (55.5% vs. 92.6%; odds ratio (OR) 0.10; 95% confidence interval (CI) 0.02-0.44). This association persisted in multivariable models after adjustment for potential confounders (adjusted OR 0.08; 95% CI 0.01-0.42; number needed to be exposed to caffeine to prevent one case of AKI = 2.6). Although baseline serum creatinine did not differ by caffeine exposure, patients receiving caffeine had lower peak creatinine (median 1.0 mg/dl vs. 1.5 mg/dl; p = 0.008) and absolute creatinine change (median 0.42 mg/dl vs. 0.68 mg/dl; p = 0.003) than those who did not. CONCLUSIONS: Caffeine exposure in preterm infants with NEC/SIP is associated with decreased incidence and severity of AKI.

Successful Treatment of IgA Vasculitis Complicated with Bowel Perforation and Crescentic Glomerulonephritis by Combination Therapy of Glucocorticoid, Cyclosporine and Factor XIII Replacement.

We report the findings of an 18-year-old boy with immunoglobulin A vasculitis (IgAV) complicated with bowel perforation and nephritis. He presented with abdominal pain, arthralgia and palpable purpura. Massive proteinuria developed during his clinical course. The patient was treated successfully using combination therapy of glucocorticoid (GC), cyclosporine (CYA) and factor XIII (F XIII) replacement. A standard treatment strategy for severe IgAV patients has not been established due to its rarity. Combination therapy using GC, CYA and F XIII replacement should be considered for severe IgAV patients.

Tratamiento conservador de perforación duodenal intraperitoneal secundaria a colangiopancreatografía retrógrada endoscópica en un paciente pediátrico / Conservative treatment of intra-peritoneal duodenal perforation after endoscopic retrograde cholangiopancreatography in a pediatric patient

Introducción: las perforaciones duodenales son consideradas lesiones de extrema gravedad a cualquier edad. Su etiología puede ser por traumatismos, o después de la realización de un procedimiento endoscópico digestivo. Debido a la elevada morbilidad y mortalidad de estas lesiones, su tratamiento quirúrgico ha sido el método de elección durante años en la mayoría de los casos. Más recientemente se ha reportado con éxito el tratamiento no quirúrgico en pacientes seleccionados. Presentación del caso: se presenta el caso de tratamiento exitoso no quirúrgico en un adolescente de 13 años con una perforación duodenal intraperitoneal después de una colangiopancreatografía retrógrada endoscópica. Las medidas terapéuticas en este caso fueron: la suspensión de la vía oral, el uso de nutrición parenteral total, la administración de análogo de la somatostatina y los antibióticos sistémicos. Conclusiones: la perforación duodenal intraperitoneal después de una colangiopancreatografía retrógrada endoscópica en niños puede ser diagnosticada precozmente. El tratamiento médico basado en la observación estricta del enfermo, la nutrición parenteral total, la suspensión de la alimentación oral, la aspiración nasogástrica octeotride y la administración de antibióticos sistémicos, es eficaz en enfermos selectos(AU)

Introduction: duodenal perforations are considered extremely serious lesions at any age. Its etiology may be due to trauma, or after performing a digestive endoscopic procedure. Due to the high morbidity and mortality of these injuries, surgical treatment has been the method of choice for years in most cases. More recently, non-surgical treatment has been reported successfully in selected patients. Case presentation: it is presented a case of successful non-surgical treatment in a 13-year-old adolescent with intra-peritoneal duodenal perforation after endoscopic retrograde cholangiopancreatography. Therapeutic measures in this case were: the suspension of the oral route, the use of total parenteral nutrition, the administration of the somatostatin analog and systemic antibiotics. Conclusions: intra-peritoneal duodenal perforation after endoscopic retrograde cholangiopancreatography in children can be diagnosed early. Medical treatment based on strict observation of the patient, total parenteral nutrition, suspension of oral feeding, nasogastric octeotride aspiration; and administration of systemic antibiotics is effective in selected patients(AU)

Malignant atrophic papulosis with motor aphasia and intestinal perforation: A case report and review of published works.

Malignant atrophic papulosis (MAP) is a rare type of obliterating vasculopathy that can present as pure cutaneous lesions or a systemic entity affecting multiple organs. Systemic disease, such as gastrointestinal or central nervous system involvement, may predispose the patients to poorer or even fatal outcomes. We present a 30-year-old female patient with systemic manifestation of MAP 10 days after delivery of a full-term pregnancy who subsequently developed motor aphasia and intestinal perforation. The patient was administrated empirical treatment with an antiplatelet, anticoagulant, methylprednisolone sodium succinate and alprostadil. Antibiotics were administrated due to intestinal perforation and secondary sepsis. Despite all treatment, the patient died a week later. We summarized all the previous reports of MAP based on thorough review of previous published work. Overall, this is the first patient with MAP combined with motor aphasia and intestinal perforation and may provide insights for future studies on the treatment of this disease.

Multiple small bowel perforations due to invasive aspergillosis in a patient with acute myeloid leukemia: case report and a systematic review of the literature.

PURPOSE: Invasive aspergillosis (IA) represents a major cause of morbidity and mortality in immunocompromised patients. Involvement of the gastrointestinal tract by Aspergillus is mostly reported as part of a disseminated infection from a primary pulmonary site and only rarely as an isolated organ infection. METHODS: We report a case of small bowel perforation due to IA in a patient with acute leukemia under chemotherapy and pulmonary aspergillosis. We performed a systematic review of the literature as well. RESULTS: A 43-year-old man with acute myeloid leukemia under chemotherapy developed severe neutropenia and pulmonary aspergillosis due to Aspergillus flavus. He developed melena and hemodynamic failure and a contrast-enhanced ultrasound scan suggested active intestinal bleeding. During emergency laparotomy we found multiple intestinal abscesses, several perforations of intestinal loop and Aspergillus flavus was isolated from the abscesses. Resection of the jejunum was performed. The patient received voriconazole and finally recovered. The patient is now alive and in complete disease remission. From literature review we found 35 intestinal IA previously published in single case reports or small case series as well. CONCLUSION: Clinical manifestations of gastrointestinal aspergillosis are nonspecific, such as abdominal pain, and only occasionally it presents as an acute abdomen. Antemortem detection of bowel involvement is rarely achieved and, only in cases of complicated gastrointestinal aspergillosis, the diagnosis is achieved thanks to the findings during surgery. Gastrointestinal aspergillosis should be suspected in patients with severe and prolonged neutropenia with or without pulmonary involvement in order to consider the right therapy and prompt surgery.

A 14-year-old girl presenting with tuberculous intestinal perforation while in a temporary shelter after the 2015 earthquake in Nepal.

A 14-year-old Nepalese girl presented with fever, abdominal pain and vomiting. She was living with her family in a temporary settlement camp following the earthquake in Nepal in 2015. She had had abdominal pain for 2 months and fever for 1 month. Abdominal examination suggested acute peritonitis. At laparotomy, three ileal perforations were detected and histopathology demonstrated caseous granulomas. Her father had sputum-positive pulmonary tuberculosis. She was diagnosed with abdominal tuberculosis and responded well to anti-tuberculosis chemotherapy. Intestinal perforation is a rare complication of tuberculosis in children.