Pneumatosis cystoides intestinalis mimicking free intraabdominal air following chemotherapy for relapsed acute myeloblastic leukemia in a transplanted neutropenic child: a case report.

BACKGROUND: Pneumatosis cystoides intestinalis (PI) is a rare but important condition in which widespread air sacs are found in the submucosa, and subserosa of the bowel wall. Although it has several etiologies, children receiving chemotherapy are at risk for PI. Preferred imaging tools for the diagnosis are abdominal direct radiography and computed tomography. In patients with PI, rupture of intramural air sacs is the source of benign pneumoperitoneum, causing free air without true intestinal perforation. Intestinal perforation or obstruction are indications for surgical intervention. CASE: Here, we present a 4-year-old patient diagnosed with acute myeloblastic leukemia (AML), who underwent allogeneic hematopoietic stem cell transplantation (HSCT) from a matched sibling donor (MSD) and developed PI after HSCT. The patient was consulted to the pediatric surgery department, and her oral feeding was stopped. Broad spectrum antibiotics (teicoplanin, metronidazol and vancomycin) were initiated. Her fever increased during the 24-hour monitoring, there was no stool passage, CRP ( > 25 mg/dL, normal value < 1 mg/dL) and abdominal distension increased and there was prolonged neutropenia and radiologic investigations could not rule out intestinal perforation, so the patient underwent exploratory laparotomy. No intestinal perforation was found. There was no sign in the intestinal wall and numerous gas-filled cysts of various sizes. CONCLUSIONS: PI is an uncommon complication, and direct radiography/computed tomography scans are very helpful in making the diagnosis in suspicious cases. PI, should be kept in mind, especially in transplanted or relapsed leukemia patients receiving intensive chemotherapy.

Prednisolone induced pneumatosis coli and pneumoperitoneum.

Pneumatosis intestinalis (PI) is defined as the presence of gas within the submucosal or subserosal layer of the gastrointestinal tract. It is a radiologic sign suspicious for bowel ischemia, hence non-viable bowel must be ruled out in patients with PI. However, up to 15% of cases with PI are not associated with bowel ischemia or acute abdomen. We described an asymptomatic patient with prednisolone-induced PI and modified the Naranjo score to aid in a surgeon's decision-making for emergency laparotomy vs non-operative management with serial assessment in patients who are immunocompromised due to long-term steroid use.

Pneumatosis cystoides intestinalis associated with etoposide in hematological malignancies: a case report and a literature review.

Pneumatosis cystoides intestinalis (PCI) is a rare condition characterized by the presence of air collection within the subserosa and/or submucosa of the gastrointestinal wall. Due to the lack of specific symptoms, PCI is likely to be misdiagnosed or missed without the use of imaging techniques or gastrointestinal endoscopy. Here, we report a patient who complained of abdominal distention and constipation after chemotherapy for hematological malignancies, and was diagnosed with secondary PCI via computed tomography (CT) and exploratory laparotomy. Pneumoperitoneum was no longer observed after two weeks of conservative treatments. Notably, the possibility of intra-abdominal pressure (IAP) as a predictor for surgical intervention was proposed. Furthermore, we conducted a literature review on PCI after chemotherapy in hematological malignancies to raise awareness of etoposide-related PCI, while whether PCI could be identified as an adverse event of etoposide requires more evidence.

Pneumatosis intestinalis associated with lenvatinib during thyroid cancer treatment: a case report.

BACKGROUND: Pneumatosis intestinalis is a rare disease characterized by gas-filled cysts within the submucosa or serosa of the intestinal tract. In recent years, pneumatosis intestinalis was reported in patients undergoing cancer treatment, and some case reports exist that report that pneumatosis intestinalis occurs during administration of vascular endothelial growth factor inhibitors, such as bevacizumab and sunitinib. Here, we report the first case of pneumatosis intestinalis during lenvatinib treatment. CASE PRESENTATION: A 77-year-old Japanese man presented to our hospital with a chief complaint of numbness in the right leg and weakness of the lower limbs 9 years after right thyroid lobectomy. Computed tomography showed a tumor 90 mm in size from the lumbar spine to the sacrum, causing spinal cord compression. Blood tests showed that the patient's thyroglobulin level was increased to 11,600 ng/ml. We diagnosed him with thyroid cancer with bone metastases. External beam radiotherapy (39 Gy/13 Fr) was performed on the bone metastases, followed by total thyroidectomy and radioactive iodine therapy. Four months after radioactive iodine therapy, lenvatinib was introduced because the symptoms of numbness and weakness recurred. Lenvatinib was introduced at dose of 24 mg, and then it was reduced to 14 mg owing to Common Terminology Criteria for Adverse Event grade 3 paronychia of the right foot. Although no further significant adverse events occurred, a scheduled computed tomography image showed pneumatosis intestinalis of the ascending colon 14 weeks after the introduction of lenvatinib. No abdominal or digestive symptoms were observed; therefore, we selected conservative treatment. We discontinued lenvatinib for a week, but we were required to restart lenvatinib as the numbness in the right leg worsened after withdrawal. Since the introduction of lenvatinib, 3 years and 5 months passed; we continued lenvatinib treatment, and the therapeutic effect remains partial response. There has been no recurrence of pneumatosis intestinalis. CONCLUSIONS: Although rare, it is important to recognize that pneumatosis intestinalis can occur in association with lenvatinib and should be differentiated from intestinal perforation. Pneumatosis intestinalis association with lenvatinib can be improved by withdrawal.

Pneumatosis intestinalis and spontaneous perforation associated with drug toxicity in oncologic patients: a case series.

Pneumatosis Intestinalis (PI) is a rare radiological finding defined as the presence of extra-luminal gas within the intestinal wall. Several anti-tumor drugs can induce a damage of the gastrointestinal walls as an adverse effect, causing loss of mucosal integrity and endoluminal gas diffusion, responsible for PI development. We retrospectively analyzed 8 cases of PI detected through radiological imaging in oncologic patients undergoing various therapeutic regimens: five patients were receiving chemotherapy, two molecular targeted therapy (MTT) and one immunotherapy. Three patients were asymptomatic and pneumatosis was incidentally detected at routinary follow-up CT and then treated conservatively. Five patients presented acute abdomen symptoms and in these cases bowel perforation was the cause of death. Our experience confirms PI and perforation as rare complications of drug toxicity, especially in oncologic patients treated with combinations of different anticancer drugs and documented the second reported case of PI associated with atezolizumab and alectinib single administration.

[Diagnosis of Serous Adenocarcinoma following Pneumatosis Intestinalis Development during Bevacizumab Therapy for Cancer of Unknown Primary-A Case Report].

A 75-year-old woman was treated with TC plus Bev for cancer of unknown primary. During treatment, she presented to the clinic with chief complaints of general malaise and anorexia. On presentation, abdominal distention and upper abdominal tenderness were noted, and sepsis was suspected. A thoracoabdominal CT scan revealed prominent intramural emphysema and mesenteric gas in the ascending colon. An emergency laparotomy was performed for suspected pneumatosis intestinalis non-obstructive intestinal ischemia. However, no intra-abdominal contamination or ischemic changes were observed intraoperatively. Histological examination revealed a small adenocarcinoma on the serous surface of the ascending colon, and immunochemical staining confirmed the diagnosis of serous adenocarcinoma as the patient's primary cancer. This report describes a case in which the patient achieved long-term survival after diagnosis. It also emphasizes the importance of identifying the subset of patients with cancer of unknown primary who have a good prognosis in order to provide appropriate treatment.

Blinatumomab-related pneumatosis intestinalis in a pediatric patient with relapsed acute lymphoblastic leukemia: A case report.

INTRODUCTION: Pneumatosis intestinalis is characterized by air in the subserosal or submucosal layer of the intestine, with the severity ranging from mild and asymptomatic to symptomatic with serious conditions such as intestinal ischemia and perforation requiring surgery. Although several etiologies, including those from conventional chemotherapy agents and molecular target agents, have been suggested, blinatumomab-related pneumatosis intestinalis is quite rare. CASE REPORT: An 11-year-old girl with history of B-cell ALL presented with bone marrow relapse 3 years after completion of initial chemotherapy. Reinduction chemotherapy and blinatumomab as post-reinduction consolidation were initiated. On day 28 of blinatumomab therapy, pneumatosis intestinalis from the ascending colon to the hepatic flexure was found incidentally on abdominal computed tomography.Management and outcome: After withholding blinatumomab therapy for 1 month, pneumatosis intestinalis improved significantly without abnormal gastrointestinal symptoms. Blinatumomab was resumed and safely completed. The computed tomography performed 4 months later showed complete resolution of pneumatosis intestinalis. The patient has been in good condition for over 1 year to date. DISCUSSION: To our knowledge, this is the first case report of pneumatosis intestinalis after blinatumomab therapy in a pediatric patient with relapsed precursor B-cell acute lymphoblastic leukemia. Herein, we highlight the importance of early detection of pneumatosis intestinalis through imaging follow-up during blinatumomab therapy.

Lactulose-induced pneumatosis intestinalis following colonoscopy: a case report.

Pneumatosis intestinalis (PI) occurs when gas is discovered in the intestinal wall and is categorized into two types: primary PI which is idiopathic and mainly occurs in the colon, and secondary PI which occurs more often in the small bowel but has variable presentation and etiology. We report a case of a patient status post-orthotopic deceased liver transplantation complicated by a portal vein thrombus on chronic lactulose for portosystemic encephalopathy who presented due to pyelonephritis and persistent diarrhea. The patient underwent colonoscopy with random biopsies and subsequently developed acute sepsis with Escherichia coli bacteremia. The findings of PI were noted on computed tomography imaging obtained 5 days post-colonoscopy, due to persistent post-procedure abdominal pain. The patient was treated with discontinuation of lactulose, supportive care, and antibiotics for her bacteremia with resolution of her PI 3 days later. This suggests that a combination of factors may lead to the development of PI, and while some cases require emergent intervention including surgery, others may be treated conservatively. Awareness of risk factors that may precipitate PI and specific clinical predictors may help to both mitigate and manage PI appropriately.

[A Case of Colorectal Cancer with Asymptomatic Idiopathic Pneumatosis Intestinalis during Bevacizumab Administration].

We present the case of a 75-year-old woman who received CapeOX plus Bmab therapy(capecitabine, oxaliplatin, and bevacizumab)after primary excision for an unresectable advanced sigmoid colon cancer with remote metastasis. Pneumatosis intestinalis(i.e., the presence of isolated gas in the abdominal cavity)was revealed accidentally during a periodical imaging examination in the small intestine and transverse colon, albeit no subjective symptoms were reported. Owing to the absence of definitive evidence of pneumatosis intestinalis and gastrointestinal perforation, the patient was diagnosed with idiopathic pneumatosis intestinalis. Bmab was discontinued, and CapeOX therapy alone was continued after follow-up. Approximately 4 months later, pneumatosis intestinalis had completely disappeared. Bmab is a vascular endothelial growth factor antibody with well-known side effect of gastrointestinal-perforation. However, there have been few reports on pneumatosis intestinalis; to our knowledge, there have been no reports on pneumatosis intestinalis associated with colorectal cancer in Japan. Further, the report suggests the need for appropriate and immediate management of pneumatosis intestinalis following diagnosis.

Pneumatosis cystoides intestinalis induced by the alpha-glucosidase inhibitor complicated from sigmoid volvulus in a diabetic patient.

INTRODUCTION: We present the case of a diabetic patient on treatment with acarbose who had presented a sigmoid volvulus with localized cystic pneumatosis of the sigmoid colon. CASE REPORT: A 72-year-old patient with a medical history of atrial fibrillation, DNID in treatment since 10 years by acarbose. The patient was admitted to the emergency for abdominal pain and occlusive syndrome since 48 h without fever or nausea or vomiting. A CT scan was performed that showed a dolichocolon with a sigmoid volvulus. The colonic wall was thickening as well as submucosal and subserosal gas, without extra digestive air or collections. A rectosigmoidoscopy was achieving that showed a sigmoid volvulus with multiple small projections like a submucosa gas bubbles. A laparoscopic non-oncologic sigmoidectomy with primary termino terminal colorectal anastomosis was performed. During the surgical procedure, an aspect of PCI of the sigmoid colon was found. The sigmoid colon was long like a dolichocolon, dilated, and partially twisted. DISCUSSION: PCI is a rare condition characterized by the presence of multiple pneumokystes at different layers of the colonic wall. In emergency setting, the presence of colonic pneumatosis precludes the differential diagnosis between the PCI and mesenteric ischemia or ischemic colitis. It can be the cause of unnecessary explorative laparotomy. CONCLUSION: PCI is rare disease, in emergency setting, we had to consider in differential diagnosis with colonic vascular disorders.

Portal vein gas and pneumatosis intestinalis: A case of intestinal necrosis caused by acute organophosphorus pesticide poisoning?

Acute organophosphorus pesticide poisoning (AOPP) is fairly common in rural areas of Asia. The symptoms of AOPP are mainly caused by acetylcholine accumulation. According to the clinical characteristics, AOPP symptoms can fall into the following three categories: muscarinic, nicotinic, and central. Death from fatal poisoning is caused by respiratory paralysis, and neurological complications are common. However, no case of intestinal necrosis caused by AOPP has been reported. Hepatic portal vein gas and pneumatosis intestinalis are considered typical and early imaging manifestations of intestinal necrosis. In this article, we describe a very rare case of computed tomography imaging-proven intestinal necrosis caused by AOPP.

Pneumatosis Intestinalis After Molecular-Targeted Therapy.

BACKGROUND: Pneumoperitoneum after surgical manipulation of the abdomen implies a perforation. Rare cases of nonoperated cancer patients, largely with gastrointestinal or genitourinary cancers, have been noted to have radiologic findings of pneumatosis intestinalis and/or pneumoperitoneum as a complication of molecular-targeted therapy (MTT) without confounding factors for perforation. We present a patient with a cranial malignancy treated with bevacizumab who subsequently manifested with pneumatosis intestinalis. CASE DESCRIPTION: A 67-year-old man with metastatic melanoma, non-small cell lung cancer, and recurrent cerebellar subependymoma was initiated on bevacizumab treatment for subependymoma recurrence. He subsequently underwent an uncomplicated ventriculoperitoneal shunt for progressive obstructive hydrocephalus, confirmed by a normal postoperative abdominal radiograph. One week later, he returned with worsening lethargy and a computed tomography consistent with pneumomediastinum and pneumoperitoneum. Due to concern for bowel perforation, the patient underwent diagnostic laparoscopy and removal of ventriculoperitoneal shunt. Focal sigmoid pneumatosis was identified without any signs of bowel perforation or ischemia. Bevacizumab was discontinued, and the patient's radiologic and clinical findings improved. CONCLUSIONS: With increasing utilization of MTTs in brain tumor management, we raise MTT as a potential cause for pneumoperitoneum in neurosurgical patients. Pneumoperitoneum after extracranial procedures still requires workup and management for potential bowel perforation, but alternative causes such as bevacizumab should also be considered. Pneumatosis intestinalis patients on MTT can have benign physical examinations and will resolve, in the majority of cases, on discontinuation of the drug.