Abstract
Milk curd syndrome is an infrequent, iatrogenic cause of bowel obstruction in neonates and early infants, which arises mainly due to inspissation of formula feeds or dried cow's milk, but is very uncommon after feeding with unmodified animal milk, such as goat's milk. A systematic review aims to gain a deeper understanding of its demographics, causes, clinical profile, diagnosis, and management. If treated promptly, outcomes are favourable.
Introduction
Obstruction from milk curd syndrome (MCS) 1 linked to unmodified animal milk, such as goat's milk, is extremely rare. 2 Affected infants usually present within 4–10 days of feeding, showing prolonged constipation and signs of small bowel obstruction. Diagnosis is typically made during surgery, with inspissated curds found in the small intestine and sometimes in the large bowel.3–5 The prognosis is generally good.
Methods
We report a series of four cases of MCS from our experience. We then performed a systematic review of the literature, following the PRISMA guidelines, and identified all published reports of MCS (also known as lactobezoar) in humans. Databases screened were PubMed/MEDLINE, PubMed Central, ScienceDirect, Wiley Online Library, and DOAJ. To capture additional references, a targeted Google Scholar search and backward citation tracking from review articles were also conducted. The protocol was not registered on PROSPERO.
We included case reports and case series published in English that described human patients diagnosed with lactobezoar or MCS. All records retrieved were imported into a reference management file. After removing duplicates, the full texts of potentially eligible studies were then assessed against the inclusion criteria, which were case reports and case series in children in the English literature but excluding non-human studies, review articles without a primary case report or series, conference abstracts without full texts, and duplicate publications.
The database searches yielded 99 records, which left 58 unique reports after removing duplicates. Following abstract and full-text review, 32 studies met our inclusion criteria and were incorporated into the qualitative synthesis.
Case series (
Table 1
)
For case series, please refer Table 1.
Case series.
Discussion
A total of 32 studies were reviewed, involving 82 patients, Table 2.
Systematic review of literature.
A higher incidence was noted in 53 males compared to 17 females (though 12 had no mention of gender). The youngest was a 3-day-old girl, and the oldest a 16-month boy. The median age (of 75 cases where age was given) was 18 days (IQR, 6–30 days), indicating a skewed distribution due to the presence of a few older children. The most frequent age at presentation was 6 days. Importantly, 60/81 infants (74.1%) presented within the first month of life, highlighting the predominance of neonatal onset.
The data on presenting symptoms, associated features, cause/type of feed, level of milk curd inspissation, investigation, and treatment modality are seen in Tables 2 and 3.
Symptomatology, cause, investigation, and management of the review cohort.
Plain abdominal radiographs and ultrasonography could not definitively diagnose MCS in all cases, but helped identify intestinal obstruction with no other attributable cause. Contrast studies were used but infrequently.
Conservative management was successful in 26/86 (30%). Depending on the site of inspissation, various non-operative modalities were used. Gastric lavage with saline or N-acetylcysteine was employed for bezoars. For more distal bowel or colonic sites, saline or Gastrografin enemas were used. Oil enema was used by one. 33 Our procedure of a single daily enema with diluted Gastrografin (1:5 in saline), followed by a saline enema at a 12-h interval. Vigilant monitoring of nasogastric output and adequate intravenous rehydration was ensured. If any rapid increase in abdominal distension or deteriorating vital signs were noted, or no improvement occurred by 48–72 h, this was deemed failed conservative management.
In such cases, if there was perforation, or if the patient was physiologically unstable, an exploratory laparotomy was performed, at which various procedures from milking or evacuation to enterotomy, clearance, or resection with ileostomy or colostomy, were carried out depending on the severity.
Demise was recorded in 9/86 patients (9.5%).
Conclusion
MCS is a rare cause of intestinal obstruction in neonates and infants. The condition has a good prognosis if managed promptly; hence, the diagnosis is imperative if without other attributable causes, but with a relevant feeding history. Radiographs and ultrasonography are crucial for diagnosis.
Footnotes
Author contribution
All the authors were involved in patient care (establishing clinical diagnosis, planning investigations, management, and follow-up), the article review process, and in writing the manuscript.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
Declaration of conflicting interests
The authors declared no potential conflicts of interest with respect to the research, authorship and/or publication of this article.