The Journal of Emergency Medicine, Vol. 48, No. 5, pp. 607–608, 2015 Copyright Ó 2015 Elsevier Inc. Printed in the USA. All rights reserved 0736-4679/$ - see front matter

http://dx.doi.org/10.1016/j.jemermed.2014.12.053

Visual Diagnosis in Emergency Medicine

PNEUMATOSIS INTESTINALIS IN A CORTICOSTEROID-DEPENDENT CHILD Andrea T. Cruz, MD, MPH,* Bindi J. Naik-Mathuria, MD,† and George S. Bisset, MD‡ *Department of Pediatrics, Baylor College of Medicine, Houston, Texas, †Department of Surgery, Baylor College of Medicine, Houston, Texas, and ‡Department of Pediatric Radiology, Baylor College of Medicine, Houston, Texas Reprint Address: Andrea T. Cruz, MD, MPH, Department of Pediatrics, Baylor College of Medicine, 6621 Fannin Street, Suite A2210, Houston, TX 77030

, Keywords—benign pneumatosis; chronic corticosteroids; pneumoperitoneum

(PI) along the ascending, descending, and transverse colon. There was no evidence of free intraperitoneal air or portal venous gas. The bowel loops were not dilated or thickened. To better evaluate this finding, computed tomography (CT) scans of the abdomen and pelvis were obtained (Figure 2). This demonstrated diffuse PI from the cecum to the distal transverse colon, without associated free fluid, pericolonic inflammatory changes, or bowel wall thickening. A normal appendix was visualized. The child was admitted to the surgical service on piperacillin/tazobactam for bowel rest and serial abdominal examinations to ensure that progression to intestinal perforation did not occur. His diet was slowly advanced after his abdominal examination remained nonconcerning for 2 days. He was discharged after a 4-day length of stay with a diagnosis of benign PI.

CASE REPORT A 12-year-old boy with pseudohypoaldosteronism, chronic lung disease, hypopituitarism, gastrostomy button, and corticosteroid-dependence presented with 1 day of crampy periumbilical and left upper-quadrant abdominal pain that did not radiate. He was afebrile and had no nausea, vomiting, diarrhea, or constipation. The family had not vented the gastrostomy tube, but he had been tolerating feeds well. On arrival, his vital signs demonstrated a temperature of 98.1 F, pulse of 86 beats/min, respiration was 24 breaths/min, blood pressure 112/71 mm Hg, and oxygen saturations of 95% in ambient air. He weighed 39 kg (28th percentile). His examination was notable for mild abdominal distension and minimal left periumbilical tenderness without rebound, guarding, or rigidity; he had normoactive bowel sounds. The remainder of his physical examination was unremarkable. Laboratory evaluation revealed a white blood count of 10,800/mL (60% neutrophils, 6% bands); bicarbonate was 24 mmol/L and lactate was 0.8 mmol/L. Aspartate and alanine aminotransferases were 33 U/L and 48 U/L, respectively. C-reactive protein was 3.1 mg/dL. Flat and upright abdominal x-rays (Figure 1) were obtained and demonstrated diffuse linear pneumatosis intestinalis

DISCUSSION PI is defined by the presence of gas in the bowel wall. PI in the pediatric age group historically has been associated with necrotizing enterocolitis, where it can represent a surgical emergency. In older children and adults, the most common causes of PI include intestinal ischemia or obstruction, thromboembolic disease, colitis, ingestions, trauma, and immunosuppression (e.g., in bone marrow transplant recipients and in patients with collagen vascular diseases) (1). In adults, pathologic PI has been associated with the need for mechanical

RECEIVED: 18 July 2014; FINAL SUBMISSION RECEIVED: 6 November 2014; ACCEPTED: 21 December 2014 607

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Figure 1. Anteroposterior abdominal x-ray demonstrated diffuse linear pneumatosis intestinalis along the ascending, transverse, and descending colon (arrows). There was no free intraperitoneal air.

ventilation, vasopressor support, renal replacement therapy, and examination findings of absent bowel sounds or clinical peritonitis (2). In children with pathologic PI, low serum bicarbonate and high serum lactate were markers of poor outcomes (3,4).

Figure 2. Computed tomography after intravenous contrast administration confirmed pneumatosis intestinalis (arrows) from the cecum to the distal transverse colon without bowel wall thickening, free peritoneal air, pericolonic inflammatory changes, or free fluid.

A. T. Cruz et al.

Benign PI has been described in patients receiving chronic corticosteroids or other immunosuppressants (5,6). In adults, the most commonly reported comorbidities associated with benign PI are pulmonary: chronic obstructive pulmonary disease, cystic fibrosis, and asthma (1). One large multicenter retrospective study estimated that 60% of patients with PI had benign PI (2). The pathophysiology of benign PI is unclear. Some experts have postulated that chronic corticosteroid use can cause atrophy of gastrointestinal lymphoid tissue, resulting in decreased submucosal integrity that may make it easier for intraluminal air to dissect into the intestinal wall (7). Patients with benign PI lack worrisome CT findings, such as bowel wall thickening or dilatation, ascites, soft-tissue stranding, free peritoneal fluid, small bowel involvement, or portal venous gas (8,9). The pattern of pneumatosis can also help distinguish benign from clinically worrisome PI. Benign PI usually is more extensive; however, the morphology of the PI (cystic or bubble-like vs. linear) did not help differentiate between the two clinical entities (9). One series of 37 nonneonates with benign PI showed that >75% had a good outcome with medical management alone (3). This child’s abdominal examination, normal laboratory evaluation, and CT findings, in conjunction with his chronic corticosteroid use, all were consistent with benign pneumatosis.

REFERENCES 1. Ho LM, Paulson EK, Thompson WM. Pneumatosis intestinalis in the adult: benign to life-threatening causes. AJR Am J Roentgenol 2007; 188:1605–13. 2. DuBose JJ, Lissauer M, Maung AA, et al. Pneumatosis Intestinalis Predictive Evaluation Study (PIPES): a multicenter epidemiologic study of the Eastern Association for the Surgery of Trauma. J Trauma Acute Care Surg 2013;75:15–23. 3. Kurbegov AC, Sondheimer JM. Pneumatosis intestinalis in nonneonatal pediatric patients. Pediatrics 2001;108:402–6. 4. Hawn MT, Canon CL, Lockhart ME, et al. Serum lactic acid determines the outcomes of CT diagnosis of pneumatosis of the gastrointestinal tract. Am Surg 2004;70:19–23. 5. Fenton LZ, Buonomo C. Benign pneumatosis in children. Pediatr Radiol 2000;30:786–93. 6. Berard R, Chedeville G, Saint-Martin C, Scuccimarri R. Benign pneumatosis intestinalis in a patient with juvenile dermatomyositis. J Rheumatol 2010;37:2442–4. 7. Heng Y, Schuffler MD, Haggitt RC, Rohrmann CA. Pneumatosis intestinalis: a review. Am J Gastroenterol 1995;90:1747–58. 8. Lee KS, Hwang S, Hurtado Ru´a SM, Jajigian YY, Gollub MJ. Distinguishing benign and life-threatening pneumatosis intestinalis in patient with cancer by CT imaging findings. AJR Am J Roentgenol 2013;200:1042–7. 9. Olson DE, Kim YW, Ying J, Donnelly LF. CT predictors for differentiating benign and clinically worrisome pneumatosis intestinalis in children beyond the neonatal period. Radiology 2009; 253:513–9.

Pneumatosis intestinalis in a corticosteroid-dependent child.

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