Milk curd syndrome, bezoar-associated neonatal stomach perforation: A case report

INTRODUCTION

Milk-related, bezoar-associated gastrointestinal (GI) obstruction in a neonate is uncommon.^[1] Milk curd syndrome (lactobezoar) is an uncommon iatrogenic form of intestinal obstruction among neonates that consist of inspissated milk aggregations. Stomach is the most common site of lactobezoar but it is a rare disorder that can also be found in the upper and lower intestine.^[2] Since 1959, only 100 cases of lactobezoar have been reported in literatures.^[3] One of the rare causes of neonatal bowel obstruction is gastric perforation.^[4]

The etiology is unknown; however, several factors include prematurity, low birth weight, thickened high-caloric formulas that contain high fat, calcium, and casein, dehydration, rapid feeding methods, and immature bowel motility. Lactobezoar can occur in newborn had been breastfed. Babies with lactobezoar present with non-specific symptoms and findings, such as abdominal distention, palpable mass, emesis, and diarrhea. Diagnosis can be made by abdominal radiographies, which may show a giant gastric radio-opaque mass like a “sandwich loaf ” or “gingerbread.” In addition, the use of abdominal ultrasound can demonstrate a giant gastric mass.^[5]

CASE REPORT

A 17-day-old newborn presented with episodes of bile vomiting, no passage of stool for 3 days, and abdominal distension. Without fever, and passed motion. The antenatal history R was unremarkable. We collected the following data for the patient: Female, weight 2800 g, 38 weeks’ gestational age, and delivered by vaginal mode. Our baby was feed high caloric preterm formula. Physical examination of the neonate was irritable, with mild dehydration and normal vital signs. Chest examination revealed good air entry and normal heart sound. Abdomen was distended and visible intestinal loops. Rectal examination was empty and normal in location. He had hypokalemia and hyponatremia.

In Figure 1, an abdominal X-ray showed pneumoperitoneum. After resuscitation, the baby was prepared for surgical intervention. Surgical exploration was done under general anesthesia, which revealed dilated intestinal loops with the presence of bowel adhesion and posterior stomach perforation [Figure 2]. Figure 3 shows that an intraluminal, inspissated, sticky, milky material like mass was extracted throughout the stomach. Peritoneal lavage was done, trimming of the perforation site, and sutured with two layers of interrupted 0-4 Vicryl. In the post-operative period, the neonate did well and woke smoothly from anesthesia admitted to the neonatal care unit, nasogastric decompression for 6 days with fluid and electrolytes resuscitation, I.V omeprazole, antibiotics, and pain killer. Oral feeding was gradually initiated and he was discharged on 7^th post-operative day. Newborn is under 3-monthly follow-up, she is gaining weight, has no abdominal distension, and passes stool daily.

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Figure 1: Anteroposterior erect X-ray of abdomen showed gasless bowel loops (Green arrow), pneumoperitoneum (Blue arrow).

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Figure 2: Intraoperative finding show posterior stomach perforation (blue arrow).

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Figure 3: Intraoperative finding show intraluminal inspissated, sticky milky material like mass was extracted throughout the stomach (blue arrow).

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DISCUSSION

Milk curd syndrome (Lactobezoar) is a rare cause of neonatal intestinal obstruction. In other series, the incidence was higher in preterm male neonates.^[6] While in our study, we report milk curd syndrome in 17-day-old, term, and female neonate who presented with greenish vomiting, not pass motion, and distended abdomen for 3 days. The peak age incidence of lactobezoar was (5–14) days of life.^[6] Our neonate was diagnosed on day 17^th during a laparotomy for neonatal pneumoperitoneum. Stomach is the most common site of lactobezoar but it is a rare disorder, only 3 neonates of stomach perforation due to inspissated gastric milk aggregations have been reported in the published literatures.^[5] Limited literatures are available on this topic; the posterior gastric wall is the most common location of stomach perforation due to lactobezoar.^[5] In lactobezoar, newborns present with a mechanical obstruction such as abdominal distention, mass, and vomiting.^[7] We suggest that the most common risk factor for developing the condition in our case was a high-calorie formula. Most newborns were premature babies in whom high-calorie feeds were initiated soon after birth.^[8] The majority of reported cases of lactobezoar were attributed to cow’s milk-based infant formula.^[6] However, with the limited literature available on this topic, it seems that the most common site of obstruction is the jejunum and ileum, but several reports of inspissated milk aggregations in the colon or even in meckel diverticulum. Sometimes, inspissated milk leads to gastric obstruction and causes posterior gastric wall perforation.^[9] In our case, the gastric lactobezoar led to posterior stomach perforation. Diagnosis of inspissated milk aggregations with GI tract obstruction can be achieved by upper GI contrast series; sometimes, other modalities are useful in diagnosis likes ultrasound.^[2] In our case, the diagnosis was done grossly during surgical exploration of neonatal pneumoperitoneum (spillage and aggregations of masses of inspissated milk at the site of stomach perforation). Treatment of lactobezoar was conservative therapy by stopping oral feedings and administer I.V fluids for few days. The use of mucolytic agent (N-acetylcystine10%) may be helpful in breaking down the lactobezoar. In other series, surgery was indicated for bowel obstruction or perforation.^[2]

CONCLUSION

High caloric formulas are the main risk factor of milk-related, bezoar-associated GI obstruction. Lactobezoar is one of the causes of neonatal bowel obstruction. Our learning outcome is to learn the mothers about the risk of high-caloric formulas and thickening of milk in the baby.