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First use of prenatal MRI to detect often-misdiagnosed CCD

Published on March 10, 2010 at 5:18 AM · No Comments

In a case believed to be a United States first, the radiology team at Lucile Packard Children's Hospital has used prenatal magnetic resonance imaging to detect an often-misdiagnosed genetic disease.

“Dr. Barth’s experience with difficult cases and the depth of his familiarity with the fetal imaging scientific literature really helped our entire team provide the best possible care for this mother and baby”

The disorder, congenital chloride diarrhea, can cause severe dehydration and serious metabolic disturbances in newborns if not treated quickly.

"This is a disease where early diagnosis is the key to a good outcome," said Richard Barth, MD, the physician who recognized the unusual case. Congenital chloride diarrhea is so rare, with only about 250 total cases reported worldwide, that infants with the disease are often erroneously treated for other diarrhea-causing ailments. "If the patient's fortunate, you could stumble onto this diagnosis," said Barth, the chief radiologist at Packard Children's and a professor of pediatric radiology at the Stanford University School of Medicine. The case was the first instance of CCD Barth had ever seen.

The diagnosis is one of only four known cases of CCD diagnosis ever made via prenatal MRI. A scientific report on the four cases, including Barth's case and three from France, was published online Dec. 9 in the journal Ultrasound in Obstetrics & Gynecology. The report is a collaboration between Barth and a team of French scientists in Marseilles.

Barth made his diagnosis in February 2009 when an expectant mom was referred to him for follow-up of an abnormal prenatal ultrasound. The ultrasound showed classic signs of bowel obstruction, a fairly common fetal problem. To get more information about the fetus, Barth ordered an MRI scan, which gave him a surprise that could not have been detected by ultrasound: Instead of showing the fetal colon filled with solid waste, as in a bowel obstruction, it was filled with fluid. This important clue, found thanks to the Packard team's expertise in fetal MRI, pointed Barth toward the obscure diagnosis of CCD. "The baby's dad said, 'You're coming up with a real zebra here,'" Barth recalls.

But that 'zebra' saved the new baby a lot of trouble when she was born at Packard a few weeks later. Barth collaborated with obstetrics and neonatology teams in the hospital’s Johnson Center for Pregnancy and Newborn Services to ensure the infant's fluid and electrolyte levels would be monitored starting at birth. This was needed because the gene mutation that causes CCD damages a salt-transporting protein in the intestine. The abnormality stops the body from absorbing essential electrolytes, causing patients to have large amounts of watery, high-salt diarrhea.

"These babies can go into shock and die because of the tremendous electrolyte imbalance at birth," said Maurice Druzin, MD, professor and service chief of obstetrics and gynecology at Packard Children's. Without the prenatal diagnosis, it could easily have taken physicians a few days to figure out what was going on, Druzin added. "The baby could have been in serious trouble by that time," he said. Untreated patients who survive early life may suffer permanent kidney damage, feeding problems, severe malnutrition, and delays in growth and motor development.

"In this case, the neonatology team was not just monitoring the baby's electrolytes, but also taking special precautions in terms of feeding," said neonatologist Susan Hintz, MD, associate professor and medical director of the Center for Comprehensive Fetal Health & Maternal and Family Care at Packard Children's. "We took these steps due to the strong possibility that this was CCD."

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