Husberg et al.

SpringerPlus (2016) 5:245

DOI 10.1186/s40064-016-1842-0

RESEARCH Open Access

Congenital intestinal malrotation

in adolescent and adult patients: a 12‑year
clinical and radiological survey
Britt Husberg1,2,3,4, Karin Salehi5,6, Trevor Peters7, Ulf Gunnarsson8, Margareta Michanek3,4,
Agneta Nordenskjöld5,6 and Karin Strigård8* 

Abstract 
Congenital intestinal malrotation is mainly detected in childhood and caused by incomplete rotation and fixation of
the intestines providing the prerequisites for life-threatening volvulus of the midgut. The objective of this study was to
evaluate a large cohort of adult patients with intestinal malrotation. Thirty-nine patients, 15–67 years, were diagnosed
and admitted to a university setting with congenital intestinal malrotation 2002–2013. The patients were divided into
three age groups for stratified evaluation. Medical charts were scrutinized, and clinical outcome of surgery was reviewed.
Twelve patients presented as emergency cases, whereas 27 were admitted as elective cases. Diagnosis was established in
33 patients who underwent radiological investigation and in the remaining 6 during surgery. A Ladd’s operation was per-
formed in 31 symptomatic patients; a conservative strategy was chosen in eight cases. Volvulus was more common in the
younger age group. Twenty-six surgically treated patients were available for telephone interview, 1–12 years after surgery.
All patients, except one, regarded their general condition improved to a high degree (n = 18) or with some reservation
(n = 7). Twelve patients suffered remaining abdominal pain of a chronic and diffuse character. Due to recurrence of malro-
tation six patients were reoperated. Symptomatic malrotation occurs in both children and the adult population. Improved
awareness and an accurately performed CT scan can reveal the malformation and enable surgical treatment. A Ladd’s
procedure relieved most patients from their severe complaints even when a history of several years of suffering existed.
Keywords:  Malrotation, Intestinal volvulus, Adult, Ladd’s procedure

Background a similar but secondary incomplete rotation and fixation

In congenital intestinal malrotation an impaired embryo- of the intestines (Torres and Ziegler 1993).
logical development of the gut causes incomplete rota- The inadequate fixation of the bowel alongside remain-
tion and fixation of the intestines to the abdominal wall ing embryonic fibrous adhesions, the Ladd’s bands (Ladd
(Dott 1923). The fulfillment of the third embryonic rota- 1932, 1936), may give rise to a variety of intestinal mal-
tion includes the traversing of the duodenum to the left function. In the worst case scenario, malrotation may
side of the abdomen, forming the ligaments of Treitz, develop into a midgut volvulus with torsion causing high
and the migration of the ileo-caecal junction to the lower risk of ischemia and necrosis of the parts of the intes-
right abdominal quadrant. The fixation of the full-length tine supplied by the superior mesenteric artery. This
bowel is complete during the twelfth week (Penco et  al. life-threatening condition is well known among pediat-
2007). Congenital malformations such as diaphragmatic ric surgeons and is always considered when physicians
hernia, omphalocele or gastroschisis are associated with treat critically ill infants with abdominal symptoms and
unknown diagnoses.
Malrotation has primarily been diagnosed in early
*Correspondence: karin.strigard@umu.se
8
Department of Surgical and Perioperative Sciences, Umeå University
childhood, with estimated onset of symptoms during the
Hospital, 901 87 Umeå, Sweden first year of life in 90  % of the cases (Vaos and Misiakos
Full list of author information is available at the end of the article

© 2016 Husberg et al. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License
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Husberg et al. SpringerPlus (2016) 5:245 Page 2 of 7

2010; Pickhardt and Bhalla 2002; Stewart et  al. 1976). de-rotated in a counter clockwise manner and all Ladd’s
There are recent reports of manifestation later in life, both bands were carefully removed and dissected. If needed,
as emergency conditions or more chronic gastrointes- the mesentery was broadened and the adhesions sur-
tinal symptoms (Penco et  al. 2007; Pickhardt and Bhalla rounding the mesenteric vessels dissected in order to
2002; Nehra and Goldstein 2011). The exact incidence of avoid future recurrence of volvulus. When the dissec-
intestinal malrotation is thus still difficult to determine. It tion was done, the small bowel was placed to the right
was earlier described to be approximately 0.2 % (Stewart and the colon to the left side of the abdominal cavity in
et al. 1976; Donnellan and Kimura 1996; Clark and Old- a “non-rotational” position. Two different surgeons reg-
ham 2002), but an incidence up to 1 % has been reported istered data from medical charts on these surgical details
(Adams and Stanton 2014). Improved radiological facili- independently.
ties, including multi-detector CT-scans, provide new pos-
sibilities to identify anatomical aberrations. Follow up
During a 12-year period, we have treated 39 consecu- The patients were routinely assessed 6  weeks, 6  months
tive cases of adult malrotation at the Karolinska Univer- and 12 months after surgery. After that, occasional con-
sity Hospital, Huddinge. The aim of this study was to tact occurred if further complaints presented. During
increase knowledge concerning this diagnosis by describ- 2012–2013 a research nurse performed telephone inter-
ing symptoms, treatment and clinical outcome in our views with a semi-structured concept concerning the
cohort of adolescent and adult patients with intestinal patients’ past and present situation and possible remain-
malrotation. ing symptoms after surgery. The questions focused on
remaining intense or chronic pain, postprandial nau-
Methods sea, vomiting and constipation. Patients were also asked
Patients whether they regarded their general physical condition as
Thirty-nine patients, 22 females and 17 males, aged improved to a high degree, improved with some reserva-
between 15 and 67 years, were diagnosed with congeni- tion or without any notable improvement.
tal intestinal malrotation. The patients were prospectively
investigated at the Karolinska University Hospital from Ethical considerations
2002 to 2013. After identification of the first patient, it The Regional Ethical Review Board approved this study
was decided to prospectively monitor patients treated for 12-06-20. Dnr 2012/957-31/3.
malrotation in order to analyze and publish data when a
reasonable number of patients had been treated. Results
Clinical data
Medical charts Twelve patients presented as emergency cases, whereas
All medical records were evaluated with regards to symp- the remaining 27 were admitted on an elective basis. The
toms, surgical procedures, previous disorders and out- most common symptom was abdominal pain, followed
comes. For analysis of differences according to age, the by signs of intestinal obstruction (Table  1). Another
patients were divided into three groups (15–20, 21–50, predominant symptom was sensations of extreme full-
51–67 years). ness and discomfort after meals, sometimes followed by
nausea and vomiting, described by 29 patients (Table 2).
Radiological diagnostics Thirteen of these patients were previously assessed and
To establish the degree of malrotation, the radiologist diagnosed with gastro-oesophageal reflux. In six cases
identified the position of the duodenum and the proximal the diagnosis was achieved during surgical treatment
small bowel, the location of the caecum and the orienta- focused on other conditions.
tion of the mesenteric vessels using intravenous, per oral Concomitant malformations were observed in 15
as well as intrarectal contrast (triple-contrast). Twisting patients (38  %), including seven patients with CNS dis-
of the mesentery of the small bowel, the “whirlpool-sign”, turbances and mental retardation. Other malformations
typical for a volvulus was noted. This evaluation was also noticed were bicorn uterus, vaginal atresia, double ure-
re-scrutinized and confirmed independently by one dedi- ters, Tuberose sclerosis, Mb Hirschprung, pelvic kidney,
cated radiologist. Cornelia de Lange syndrome and scoliosis. Eight patients
had a history of disease within the hepatobiliary and pan-
Surgery creatic system with a history of pancreas divisum and in
Symptomatic malrotation was treated by corrective sur- four cases pancreatitis. Six further patients had gastro-
gery according to the technique originally described intestinal motility disturbances, verified by small bowel
by Ladd. If a volvulus was present, the intestines were manometry and/or full thickness specimens.
Husberg et al. SpringerPlus (2016) 5:245 Page 3 of 7

Table 1  Clinical data

Total Age <21 years Age 21–50 years Age >50 years

Sex ratio (m:f ) 17:22 5:5 5:13 6:4

Number patients 39 10 18 11
Secondary malrotationa 3 1 2 0
Symptoms at diagnosis
 Abdominal pain 31 (79 %) 7 (70 %) 16 (89 %) 8 (73 %)
 Intestinal obstruction 5 (13 %) 3 (30 %) 1 (6 %) 1 (9 %)
 Incidental diagnosis 3 (8 %) 0 (0 %) 2 (11 %) 1 (9 %)
Duration of symptoms
 Hours/days 3 (8 %) 1 (10 %) 1 (6 %) 1 (9 %)
 Months 7 (18 %) 1 (10 %) 1 (6 %) 5 (45 %)
 Years 26 (67 %) 8 (80 %) 13 (72 %) 5 (45 %)
 During childhood 19 (49 %) 6 (60 %) 10 (56 %) 3 (27 %)
Imagingb
 UGI 4 2 0 2
 CT 32 7 16 9
 MRI 1 0 1 0
 “Whirlpool sign”c 7/33 (21 %) 1/5 (20 %) 3/13 (23 %) 3/7 (43 %)
Treatment
Conservative treatment 8 (21 %) 0 (0 %) 4 (22 %) 4 (36 %)
Ladd’s surg. procedure 31 (79 %) 10 (100 %) 14 (78 %) 7 (64 %)
Midgut volvulus without impaired bloodflow 7 1 5 1
Midgut volvulus with impaired blood flow 8 5 1 2
 Resection small intestine 4 3 0 1
Recurrencies 5 (16 %) 2 (20 %) 2 (14 %) 2 (29 %)
a
  CDH n = 1, gastroschisis n = 1, omphalocele n = 1
b
  “Imaging” denotes the radiologic procedure that lead to diagnosis. Two patients had no imaging due to emergency surgery (Age ≤20 years n = 1, age 21–50 years
n = 1)
c
  Out of 33 patients where CT-studies were available for reviewing

Table 2  Preoperative symptoms from medical charts (n = 31) and postoperative symptoms from a telephone interview
(n = 26)
Total Age <21 years Age 21–50 years Age >50 years

Symptoms preop (one or more symptoms from medical charts, n = 31)

 Number 31 10 14 7
 Fullness after meals 25 (81 %) 7 (70 %) 14 (100 %) 4 (57 %)
 Pain 29 (94 %) 9 (90 %) 13 (93 %) 7 (100 %)
 Constipation 13 (42 %) 4 (40 %) 7 (50 %) 2 (29 %)
Symptoms postoperative (from a telephone interview, n = 26)
 Number 26 5 14 7
 Free of symptoms 10 (38 %) 2 (40 %) 4 (29 %) 4 (57 %)
 Fullness after meals 8 (31 %) 1 (20 %) 5 (36 %) 2 (29 %)
 Pain (chronic) 12 (46 %) 3 (60 %) 6 (43 %) 3 (43 %)
 Pain (“malrotation-like”) 1 (4 %) 1 (20 %) 0 (0 %) 0 (0 %)
 Constipation 8 (31 %) 0 (0 %) 8 (57 %) 0 (0 %)
 Symptoms postoperative 16 (62 %) 3 (60 %) 10 (71 %) 3 (43 %)
 Improved QoL 25 (96 %) 4 (80 %) 14 (100 %) 7 (100 %)
Husberg et al. SpringerPlus (2016) 5:245 Page 4 of 7

Radiological findings
Investigation with multi-detector computer tomography
was used in 32 cases and MRI in one. The three cardi-
nal radiological diagnostic criteria were identified in
19 of the cases. In all but one of the patients, the small
bowel was located to the right with a pathological ver-
tical course of the duodenum that failed to traverse the
vertebral spine. In the remaining patient, the duodenal
course initially crossed the midline to the left, but turned
back again forming a loop. In addition, the ascending
colon had a short attachment to the parietal left side. In
22 cases the caecum had the expected abnormal posi-
tion according to radiology, whereas the caecum in the
remaining 11 patients was located on the right side. It Fig. 2  CT scan in axial position showing the inverted vessels
was later revealed during surgery that in all these cases
the ascending colon was mobile and not fixed to the pari-
etal abdominal wall, except in one case where the right
flexure of colon was shortly attached. Malposition of the
superior mesenteric artery and vein was noted radiologi-
cally in 26 patients, of whom 25 had inverted vessels and
one presented vessels in a vertical position (Figs. 1, 2). A
“whirlpool-sign” signifying a presence of rotation of the
bowel could be detected in seven cases (Fig. 3).

Fig. 3  Whirl pool sign where when the mesenteric of the small
intestine has been twisted

Surgery
Thirty-one patients were operated (Fig.  4). Sixteen
patients had undergone previous abdominal surgery
before the Ladd procedure, with chart notifications of
intestinal malrotation in 11 of the cases. Emergency sur-
gery was performed in 9 of 31 cases. In three patients
the operation was performed semi-urgently because of
progression of abdominal complaints. One patient had a
complete mid-gut volvulus causing ischemia and necro-
sis of the bowel, necessitating resection of the entire
small bowel. Another seven patients exhibited signs of
acute volvulus, compromising circulation in a segment
of the small bowel (2 of them >50 years). Three of these
required minor resections. There was a tendency towards
an increased risk for volvulus in the younger patients
(Table 1).
Seventeen symptomatic patients were operated on elec-
Fig. 1  CT scan showing inverted vessels, front view
tively after a radiological diagnosis. Appendectomy was
Husberg et al. SpringerPlus (2016) 5:245 Page 5 of 7

Postoperative clinical outcome

Twenty-seven patients had an uneventful postopera-
tive course, leaving the hospital within a week. Three
patients had a prolonged hospital stay due to transient
postoperative intestinal failure and one died shortly
postoperatively in the aftermath of midgut volvulus with
total bowel necrosis. An early routine follow-up after
6  weeks confirmed that all patients except one were
relieved from episodes of intense abdominal pain. Care-
takers of the mentally disabled patients stated that their
patients exhibited less signs of distress from episodes of
pain. Clinical and/or radiological signs of late recurrence
appeared in six patients requiring surgery once or twice.
Surgical procedures are described in a flowchart (Fig. 6).

Mortality
Altogether five patients operated for congenital malrota-
tion have died through the course of this study. Four of
them died due to co-morbidity not related to the malro-
tation syndrome or surgery. These patients had all under-
gone a follow-up CT without signs of recurrence.

Telephone interview
Twenty-six patients were available for a telephone inter-
view and were asked about their situation after surgery.
Fig. 4  Ladds band to be dissected and removed Details are shown in Table 2.

Discussion
performed in all cases where the appendix still remained, Intestinal congenital malrotation should be recognized
in order to avoid future diagnostic problems caused by as a reason for abdominal pain also in adults which has
the new position of the intestines in the abdomen. In 27 also been emphasized in a recent population based study
patients twisting of the mesentery between 1 and 3 turns by Coe et  al. (2015). We describe a substantial number
was described at the initiation of surgery (Fig. 5). of symptomatic patients being diagnosed in mature age
Eight patients chose a conservative attitude awaiting often after several years of suffering. Malrotation may
eventual more disabling symptoms, an attitude that was present with alarming symptoms, causing life-threaten-
more common in the older patients. ing conditions which in one case resulted in death due to
short bowel syndrome. We also show that young adults
have a tendency towards more severe symptoms requir-
ing emergency treatment. No statistical comparison
has been made with patients suffering adhesive bowel
obstruction from other reasons, and thus age and con-
current developmental disorders are the only markers
identified necessitating increased awareness when con-
sidering malrotation as cause for obstruction with severe
symptoms.
In this 12-year clinical study, the majority of the
patients experienced a considerable improvement in
their general status after surgical intervention. Nehra
presents an excellent retrospective study, which includes
130 patients of all ages treated at a single institution
(Nehra and Goldstein 2011). Only 30  % of the patients
were below 1 year of age, and as many as 48 % were above
Fig. 5  Twisting of the small bowel 18 years of age at the time of diagnosis. They described a
Husberg et al. SpringerPlus (2016) 5:245 Page 6 of 7

Fig. 6  Flowchart over the procedure for the patients

decreased risk for volvulus with age, which also was con- focusing on appropriate radiological signs provides at
firmed among adult cases in the present study. Conse- least a suspected diagnosis, while waiting further assess-
quently, a conservative attitude towards surgery is more ment. In children a contrast enema of the stomach and
reasonable in the older age group. small intestine is usually enough to diagnose malrotation
The increased recognition of intestinal malrotation where the displacement of duodenum is clearly shown. In
in adults may be explained by the more frequent use of adults, where other reasons for intestinal obstruction are
abdominal CT-scan and refinements of methods that more frequent, a more detailed imaging including exact
more correctly visualize variations in the abdominal criteria prior to surgery is valuable.
anatomy (Pickhardt and Bhalla 2002; Emanuva et  al. One third of the patients were operated as emergen-
2011). A multi-detector CT-scan provides the possibility cies, compared to the higher incidence of 75 % reported
of following the exact course of the duodenum as well as in pediatric series (El-Gohary et al. 2010). Many patients
the position of the small bowel and the caecum. Impor- had ongoing abdominal discomforts since childhood,
tantly, the orientation of the superior mesenteric vessels while others encountered a relatively sudden onset of
also becomes assessable, sometimes with an additional symptoms leading to chronic episodes of abdominal pain.
depicted rotation of the mesentery of the bowel forming A considerable proportion of the patients in this series
a “whirlpool-sign”. This may indicate a precarious circula- had reached a high age before being informed of their
tion of the bowel, possibly requiring rapid surgical inter- abnormality. Gastroenterologists and surgeons treating
vention. In the present study there were 11 patients with adults probably put less emphasis on the possibility of a
the ascending colon located at an allured right abdominal congenital malformation causing the abdominal symp-
quadrant. This confirms that a “normal-looking” anatomi- toms (Nehra and Goldstein 2011; Nagdeve et al. 2012).
cal finding of the colon should not rule out the malrotation Intestinal malrotation may have a “syndromal” appear-
diagnosis as earlier reported by El-Gohary who describes ance and is often accompanied by other anomalies (30–
reciprocal findings in 20 % of the cases (El-Gohary et al. 80  %), including developmental disorders of the CNS
2010). The entire set of radiological criteria was demon- (Penco et al. 2007; Nagdeve et al. 2012). It is important to
strated in only 18 of the investigated patients. However, all have a vigilant strategy for malrotation when investigat-
patients exhibited at least one of the radiological criteria ing abdominal complaints in mentally disabled patients
for malrotation, implying that a radiological investigation who lack the possibility to describe their symptoms. The
Husberg et al. SpringerPlus (2016) 5:245 Page 7 of 7

comorbidity caused by these concurrent disorders may Sweden. 7 Department of Radiology, Karolinska University Hospital, Stockholm,
Sweden. 8 Department of Surgical and Perioperative Sciences, Umeå Univer-
be one reason for the high mortality during follow up sity Hospital, 901 87 Umeå, Sweden.
(5/39), since only one patient died from complications
after surgery in terms of short bowel after resection of Acknowledgements
The Swedish Research Council, the Foundation Frimurare Barnhuset Stock-
ischemic intestine. holm, the Stockholm City Council, the Swedish Society for Medical Research
In pediatric reports, the recurrence rate after Ladd’s and Karolinska Institutet supported this work.
procedure is considered low with a reported incidence
Competing interests
between 2 and 7 % (El-Gohary et al. 2010; Freitz and Vos The authors declare that they have no competing interests.
1997). The higher recurrence rate reported here is partly
explained by a learning curve among the involved sur- Received: 10 November 2015 Accepted: 15 February 2016
geons, but more long-lasting preoperative symptoms may
also add to the complexity of surgical problems. Interest-
ingly, it has been shown that also children operated later
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