TO THE EDITOR: A 21-year old male patient was referred from another hospital for recurrent monthly episodes of abdominal pain of 2-3 hours duration, usually during fasting. These symptoms started about 2 years before the admission, and in a couple of occasions plain abdominal films raised a suspicion of intestinal occlusion. Fortunately, all episode resolved spontaneously or after spasmolytic/benzodiazepine therapy, and no surgical approach was attempted. No defecatory abnormalities were reported, and no secondary causes (neurological, endocrine, metabolic, and neoplastic) for the symptoms’ onset were detected. Upper panendoscopy showed the presence of duodenal erosions. The patient was therefore referred to evaluate upper gastrointestinal motility for a suspicion of chronic intestinal pseudo-obstruction (CIPO). A 6-hour fasting (since the patient refused to eat during the examination) antroduodenojejunal motility study was carried out according to a previously published technique.^1,2 Analysis of the tracing showed the presence of duodenojejunal bursts of contractions (defined as periods of > 2 minute duration with continuous high amplitude [> 20 mmHg] and high frequency [10-12/minute] phasic pressure activity that were not propagated and not followed by motor quiescence)² with slight raising of the baseline, and prolonged periods of discrete clustered contractions (defined as groups of phasic waves with amplitude > 15 mmHg and duration of 1 minute, often with some tonic elevation, preceded and followed by at least 30 seconds of quiescence)²; these findings were associated with abdominal pain similar to that complained at home. Only one normal activity front was recorded, just before stopping the recording (Figure). Although non-specific (since it may be recorded in several gut pathologic conditions including CIPO, irritable bowel syndrome, celiac disease, food allergy, etc^3,4), the above abnormalities were suggestive of neuropathic motor abnormalities of the upper gut. Since no radiological anatomic investigations of the gut had been done, due to the young patient’s age, we suggested the referring colleagues to perform a barium study. This was subsequently carried out, and showed delayed gastric emptying and abnormal rotation of the small bowel, suggesting the presence of mesenterium commune. This condition was discussed with the patient, and a surgical correction (Ladd’s procedure) was carried out, without complications. The post-operative period was uneventful, and he enjoyed good health in the following years, with yearly telephone contacts until four years ago, when it was lost at follow-up due to relocation in another city for employment change.

Figure 1. (A) Duodenojejunal manometric tracing showing the presence of a burst of contractions (asterisk, sixth tracing) and of discrete clustered contractions (last tracing). The first 5 recording points (spaced 2 cm apart) are in the antrum and proximal duodenum, and the last 2 (spaced 12 cm apart) in the distal duodenum and jejunum. (B) Another recording period showing the presence of a burst of contractions (asterisk) and of discrete clustered contractions (arrow). Recording points as in (A). (C) Only this trace, obtained just before the end of the session, shows the normally propagated activity front during a 6-hour fasting period.

When the mesentery does not rotate during embryonic development, mesenteric attachment is lacking, leading to an abnormal adult conformation known as malrotation.⁵ This malrotation represents one of the major causes of fatality in infants¹ and may display several variants, including the so-called mesenterium commune. Although the latter is usually observed in children and be complicated by a volvulus,⁶ there are sporadic reports of cases in childhood or adult patients in whom the diagnosis is made after a relatively long period of chronic or recurrent aspecific abdominal complaints.^7,8 However, to the best of our knowledge there are no literature studies evaluating gut motility in these patients. As often reported in adults,⁸ the complained upper abdominal symptoms were chronic and recurrent, and the referring physicians suspected the presence of an underlying CIPO. Although the manometric findings were non-specific, there was correlation between the motor abnormalities and the patient’s symptoms, suggesting that the intestinal malrotation might have somewhat influenced upper gut motility. This hypothesis was enforced by the fact that the symptoms completely resolved after surgical correction, and did not reappear during a long clinical follow-up. As recently reported, the Ladd’s procedure (division of Ladd’s band, lengthening of the duodenum, widening of the mesentery, and derotation of midgut) is the most common surgical approach carried out in these patients.⁹

In conclusion, manometric investigations may reveal of clinical utility in identifying the possible presence of functional abnormalities responsible for upper abdominal symptoms in selected patients. The recent availability of high-resolution intestinal manometry could also help in a better identification of these abnormalities.¹⁰

The patient’s written consent was obtained for publication of this case.

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Gabrio Bassotti and Roberto Cirocchi cared for the patient; Gabrio Bassotti carried out manometric examination and wrote the draft of the manuscript; Roberto Cirocchi contributed to the revision of the text; and both authors revised and approved the final version of the manuscript.