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Is pneumatosis cystoides intestinalis a lymphatic pathology?: A case of small bowel lymphangioma and subsequent development of pneumatosis cystoides intestinalis in a 57-year-old female.

ABSTRACT: The etiology of pneumatosis cystoides intestinalis (PCI) is multifactorial and its corresponding treatment is similarly complex. One causation of PCI, is lymphatic disruption. This association is discussed in this case report involving an incident of abdominal lymphangioma and subsequent development of PCI in a 57-year-old female. The purpose of this paper is to further understand the pathology of PCI, specifically as it relates to lymphatic disruption. Furthermore, the purpose of this article is to define a treatment protocol for this disease when it involves lymphatic pathology; i.e. when a surgical vs conservative approach is warranted.

SUBMITTER: Harris E

PROVIDER: S-EPMC6344922 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:ObjectivesPneumatosis intestinalis is a rare condition with subserosal or submucosal gas-filled cysts of the gastrointestinal tract. It is often associated with acute mesenteric ischemia, but also non-ischemic causes are described.Case presentationA 27-year-old male patient with severe congenital spastic tetraparesis presented to the emergency room with fever and reduced general condition. The patient was hypotonic and tachycardic, had a fever up to 39.7 °C and reduced peripheral oxygen saturation. The laboratory analyses revealed leukocytosis (16.7 G/L) and elevated CRP (162 mg/L).The patient was admitted to the intensive care unit (ICU) for invasive ventilator treatment because of global respiratory insufficiency and antibiotic therapy due to acute pneumonia and severe acute respiratory distress syndrome (ARDS). In addition, he suffered from colonic pseudo-obstruction but with persistent stool passage. After pulmonary recovery, he was transferred to the normal ward of internal medicine, but signs of colonic pseudo-obstruction were still present.Under therapy with diatrizoic acid and neostigmine, the abdomen was less distended, and the patient had regular bowel movements. After four days, the patient developed sudden acute abdominal pain and suffered sudden pulseless electrical activity. Immediate cardiopulmonary resuscitation was provided. After the return of spontaneous circulation, the patient underwent computed tomography (CT) and was re-admitted to the ICU. The CT scan showed massive dilatation of the colon, including pneumatosis coli, extensive gas formation within the mesenteric veins and arteries, including massive portal gas in the liver, the splenic vein, the renal veins, and disruption of abdominal aortic perfusion. The patient was then first presented for surgical evaluation, but due to futile prognosis, treatment was ceased on the ICU.ConclusionsIn conclusion, colonic pseudo-obstruction might have led to colonic necrosis and consecutive massive gas formation within the mesenteric vessels. Therefore, intestinal passage should be restored as soon as possible to avoid possible mortality.

Project description:Pneumatosis cystoides intestinalis (PCI) is characterized by gas-filled cysts within gastrointestinal tract wall from esophagus to rectum, with preferential involvement of large and small intestine. PCI is rare with an estimated incidence of 0.03 to 0-2% in general population. PCI can be distinguished into idiopathic (15%) or secondary (85%) and the clinical picture ranges from completely asymptomatic to life-threatening intraabdominal complications. Although etiology of PCI appears to be multifactorial, the exact pathophysiology is poorly understood and two main theories have been proposed (mechanical and bacterial). Over the last decades, an enormous therapeutic armamentarium was considered in PCI's management, including hyperbaric oxygen therapy (HBOT). Treatment comprises conservative treatment in mild cases to surgery in highly symptomatic and complicated PCI. In the late 70s, HBOT started to be used in selected cases of PCI not responding to conservative measures. Since then, several case reports, case series, and reviews have been published in the literature with variable outcomes. The overall response rate and complete response were 92.1% (n = 82/89) and 65.2% (n = 58/89), respectively, with a median follow-up of 7 months. Furthermore, HBOT is extremely safe, with few reported complications in the literature when used for PCI. Nevertheless, a randomized, controlled, and double-blind clinical trial is unlikely to occur given the rarity of PCI, logistical issues of HBOT, and methodological considerations related to adequate blinding with a sham-controlled group. HBOT in combination with personalized diet and antibiotics may be beneficial for moderate to severe PCI in patients with no indication for emergency exploratory laparotomy. The purpose of this article is to synthesize the existing data, analyse results of previous studies, identify gaps in knowledge, and discuss PCI' management, including the proposal of an algorithm, with a special focus on HBOT.

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Is pneumatosis cystoides intestinalis a lymphatic pathology?: A case of small bowel lymphangioma and subsequent development of pneumatosis cystoides intestinalis in a 57-year-old female.

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