Pneumatosis intestinalis during chemotherapy with nilotinib in a patient with chronic myeloid leukemia who tested positive for anti-topoisomerase I antibodies.

A 55-year-old man with several comorbidities including idiopathic interstitial pneumonia under long-term corticosteroid therapy, longstanding myocardial infarction, chronic heart failure, paroxysmal atrial fibrillation, gastro-esophageal reflux disease, constipation, and history of paralytic ileus, was diagnosed with chronic myelogenous leukemia (CML) in the chronic phase. He also tested positive for anti-topoisomerase I antibodies without clinical diagnosis of any connective tissue disease, including systemic sclerosis. Approximately 5 months after the initiation of nilotinib for CML, he developed upper abdominal distension with intermitting abdominal pain, and based on abdominal computed tomography findings, a diagnosis of pneumatosis intestinalis (PI) was made. Five courses of hyperbaric oxygen therapy quickly eliminated the PI and related symptoms without the cessation of nilotinib and, thereafter, additional oral prokinetic agents and non-absorbable antibiotics ensured the non-recurrence of PI. At 6 and 18 months after commencing nilotinib therapy, major and complete molecular response were achieved, respectively. It is suspected that both gastrointestinal hypokinesis related to the presence of anti-topoisomerase I antibodies and mucosal permeability due to corticosteroid therapy had existed. Thus, subsequent administration of nilotinib may have triggered PI by depressing gastrointestinal motility via the inhibition of c-kit.

[Pneumatosis cystoides intestinalis]. / Pneumatosis cystica intestini.

Authors present a case history of pneumatosis cystoides intestinalis with free intraperitoneal gas mimicking abdominal hollow organ perforation. The illness occurred in old immunocompromised man. The diagnosis was based on exploratory laparotomy and supported by abdominal x-ray and ultrasound.

Pneumatosis intestinalis in an infant undergoing bone marrow transplantation for Wiskott-Aldrich syndrome.

A 7-month-old patient with Wiskott-Aldrich syndrome (WAS) developed pneumatosis intestinalis (PI) in the immediate post-transplant period after receiving paternal human leucocyte antigen (HLA) phenotypically matched bone marrow (BM). PI has been described in patients with congenital or acquired immunodeficiency states and after bone marrow transplantation (BMT). To our knowledge, the condition has not been described in WAS. The underlying bowel mucosa damage as a result of the history of massive rectal bleeding, the effects of the conditioning regimen, immunosuppression, neutropenia, and infection, may all have contributed to the development of PI. Although the condition resolved by conservative management alone, the patient developed Klebsiella pneumonia sepsis, interstitial pneumonitis, failed to engraft, and died on day +66 following a second infusion of stem cells mobilized from his father's peripheral blood.