Duodenal Obstruction Caused by Acute Appendicitis with Intestinal Malrotation in an Adult. --A Case Report-Hiroaki UEO*, Masaaki NAGAMATSU*,Akira NAKAMURA**,Ryuji MATSUURA** and Osami HARA*** ABSTRACT: A 55 year old man with a short history of c o n t i n u o u s vomiting and recent episodes of midabdominal pain and high fever was discovered to have a complete duodenal obstruction caused by acute appendicitis and intestinal malrotation. A fibrous adhesion caused by the inflamed appendix in the high caecum involved the duodeno-jejunal junction. This case is unique in that the onset of acute appendicitis triggered duodenal obstruction in the presence o f an asymptomatic malrotation. KEY WORDS: rotation
duodenal obstruction, acute appendicitis, intestinal mal-
company malrotation. We report herein an INTRODUCTION
D u o d e n a l obstruction secondary to intestinal malrotation is not so rare in neonates and young children but is quite rare in adults. 1-3 T h e related complication seen in adults is obstructions of the small intestine resulting from a midgut volvulus 3-9 and most documentated cases of duodenal obstruction are related to either midgut malrotafionS,4,s, 9 or peritoneal bands.l~ 11 Making an immediate diagnosis of acute abdomen in asymptomatic adult malrotation can be difficult if the physician has limited experience with the various types o f complications that may ac-
*The Department of Surgery, Medical Institute of Bioregutation, Kyushu University,Japan **The Department of Surgery, Oita National Hospital, Oita,Japan ***The Departmentof Gastroenterology, Oita National Hospital, Oita,Japan Reprint requests to: Hiroaki Ueo, MD, The Department of Surgery, Medical Institute of Bioregulation, Kyushu University69, 4546 Tsurumihara, Beppu 874, Japan
unusual case of complete duodenal obstruc-
tion caused by acute appendicitis in a 55 year old man. A CASE REPORT A 55 year old m a n was admitted to our department on January 4, 1988, with complaints of continuous nausea and recent episodes o f severe midabdominal pain and high fever. Eight days prior to his admission, he experienced severe midabdominal pain in the area of the umbilicus. Two days later, a gastrofiberscopic examination showed no abnormal findings in the stomach or duodenal bulbus, after which the abdominal pain diminished spontaneously for 2 days then re-occurred in conjunction with abdominal tenderness and a body temperature of over 39~ He visited his physician again and was prescribed antibiotics for the next 3 days, however, the abdominal pain and fever persisted and he began to complain of nausea and vomiting on the third day of this treatment. T h e patient was then transferred
JAPANESEJOURNALOFSURGERY,VOL.20, No. 3 pp. 346-350, 1990
Volume 20 Number 3
347
Duodenal obstruction caused by appendicitis
Fig. 2. Barium meal study. The duodenal mucosa at the obstruction (see arrow) was normal, suggesting compression by the surrounding tissues.
Fig. 1. Plain X-ray film revealed enlargement of the stomach and duodenum. Since the position of the duodenum appeared to be normal, intestinal malrotation was not diagnosed. to our hospital for detailed investigations. On admission, the patient had no abdominal pain, abdominal distention or fever but had been constipated for 3 days. A nasogastric tube was inserted for gastric and bile juice collection. Vomiting was controlled by fasting and total parenteral nutrition was commenced. Physical examination revealed clinical dehydration and slight distention in the upper region of the umbilicus but no rebound tenderness. A leukocytosis o f ll,200/mm 3 suggested an inflammatory focus. The erythrocyte sedimentation rate was 15/36 mm and c-reative protein was 3.36 u/ml. A plain X-ray film showed dilatation of the stomach and d u o d e n u m (Fig. 1). A barium meal revealed obstruction at the duodeno-jejunal junction seemingly caused by a compression by the surrounding tissues rather than by a duodenal mucosal lesion (Fig. 2). Computed tomography showed no evidence of a low density area in the pancreas or retroperitoneum. A barium e n e m a revealed the caecum to be located in a slightly higher and inner position than nor-
mal and the appendix was not clearly evident (Fig. 3A). Nevertheless, a diagnosis o f intestinal malrotation and acute appendicitis was not made. Since the amount o f gastric and bile juice aspirate increased progressively, a laparotomy was performed on january 25, 1988, which revealed a complete malrotation, and showed the caecum and fight colon to be mobile and high, and advanced appendicitis. An abnormal mesenteric fixation was also present and fibrous adhesion caused by the inflamed appendix involved the duodenojejunal junction at the mesenteric pouch (Fig. 4). Ladd's band was present but was not the cause o f the duodenal obstruction. With appendectomy and release o f the adhesion, the duodenal obstruction was completely reduced (Fig. 3B). T h e patient's postoperative course was uneventful and he has had no further complaints over the subsequent 15 months. Histological examination showed acute inflammatory infiltration in the appendix wall and no malignancy. DiscussioN Most symptomatic anomalies of intestinal r o t a t i o n are detected in the n e o n a t a l period. 1,~ In fact, Kiesewetter and Smith = stated that half the n u m b e r o f such cases are diagnosed during the first postnatal week
Ueo et al.
348
JpnjviJy Surg. 1990
Fig. 3. A Barium enema showing a mobile caecum and poorly identified appendix (arrows). B Postoperative barium meal study.
Fig. 4. IUustration of malrotation and inflamed appendix obstructing the duodenojejunal junction. and eighty per cent within the first month. O n the other hand, most occurrences in
adults are asymptomatic and some are incidentally discovered at routine screenings or during a laparotomy for other disorders. The most frequent symptomatic presentation in adults is intestinal obstruction with midgut volvulus. G a r d n e r and H a r t a reviewed 105 cases of intestinal obstruction caused by abnormalities o f intestinal rotation and stated that 88 were caused by a volvulus of the entire mesentery, 10 by obstruction of the transverse colon and 7 by obstruction o f the d u o d e n u m by a b n o r m a l intestinal fixation. Delvin 4 reported 7 adult cases o f midgut malrotation causing intestinal obstruction and Cathcart et al. ~ reported 5 adult patients with midgut nonrotation. Balthazar 6 assessed roentgenographic presentations in 28 adult cases and noted two different patterns, one b e i n g the classical f o r m of nonrotation of the entire midgut and the other, the isolated form o f complete a n d partial rotation. In most of these reported cases of midgut malrotation in adults, however, abdominal disorders h a d b e e n p r e s e n t since childh o o d ? -~ Two cases of midgut volvulus first
Volume 20 Number 3
Duodenal obstruction caused by appendicitis
349
TaMe 1. Cases of Duodenal Obstruction with Anomalies of Intestinal Rotation Reporters
Total Cases
Duodenal Obstruction
Midgut malrotation
Gardner and Hart s Delvin4 Van Wingerden and Dons 8 FisherJ.K. 9
46 7 1 1
6 2 1 1
Abnormal intestinal fixation
Gardner and Hart s
2
2
Peritoneal bands (Ladd's band)
Ellenberg and Delcastillo 1~ Ichimura T. et alY
1" 1
1" 1
Acute appendicitis
Ueo et al. (the present case)
1
1
Cause
*; a 10 year old child; all others were over the age of 14 years.
occurring in middle age were reported by Rowson et al., 7 and our patient h a d also b e e n symptom-free until the acute onset o f appendicitis caused the duodenal obstruction. The documented cases of duodenal obstruction occurring in adults and adolescents with anomalies of intestinal rotation are summarized in Table 1. Duodenal obstruction is usually related to midgut malrotation Or p e r i t o n e a l b a n d s . I n o n e series 3 o f anomalies o f intestinal rotation, 17 o f 88 patients with midgut malrotation h a d a duodenal obstruction. Moreover, in the same report, there were 7 cases o f duodenal obstruction caused by a b n o r m a l intestinal fixation, 2 of w h o m were adults. Van Wingerden and Dons 8 reported complete duodenal obstruction in a 24 year old p r e g n a n t w o m a n with intestinal malrotation and midgut volvulus. Ellenberg a n d Delcastillo 1~ reported a case of duodenal obstruction from peritoneal (Ladd's) bands and I c h i m u r a et alY treated an adolescent with duodenal stenosis caused by Ladd's bands and intestinal malrotation. However, our study of the literature revealed no documentation o f duodenal obstruction caused by acute appendicitis with intestinal malrotation. As for appendicitis causing intestinal obstruction in the case o f malrotation in adults, I n g r a m and Garnder 12 reported one case o f a n obstruction o f the ileum in the left iliac fossa, in the presence of
an inflamed appendix. B e c a u s e the caecum is often located in the u p p e r portion around the d u o d e n u m in patients with malrotation and because acute appendicitis is c o m m o n in adults, duodenal obstruction from acute appendicitis may occur more frequently than expected. O n e pertinent clinical indication of duodenal obstruction is a copious a m o u n t of bile-stained vomitus. Plain X-ray film and barium meal study clearly revealed a dilated duodenal loop and the findings o f the barium e n e m a supported the diagnosis of intestinal malrotation with a mobile caecum. A poorly identified appendix on the barium e n e m a in c o n j u n c t i o n with t e n d e r n e s s around the mobile caecum also suggested an inflamed appendix. W h e n patients with vomiting caused by duodenal obstruction have tenderness in the u p p e r region o f the umbilicus, the possibility of acute appendicitis with intestinal malrotation should be considered, especially in patients with previous episodes o f gastrointestinal complaints. T h e surgical treatment involves releasing the adhesion which obstructs the d u o d e n u m and removing the appendix. T h e intestine is then returned to its normal position and no particular fixation is required. Stauffer and H a r r m a n n 13 c o m p a r e d the late results of 77 surgical patients with intestinal malrotation who underwent or did not undergo additional intestinal fixation. T h e y found that
350
Ueo et al.
intestinal fixation reduced neither the incidence of reoperation nor that of postoperative abdominal complaints. (Received for publication onJun. 19, 1989)
7.
8.
REFERENCES 1. Stewart DR, Colondy AL, Daggett WC. Malrotation of the bowel in infants and children: A 15 year review. Surg 1976; 79: 716-720. 2. Kiesewetter WB, Smith JW. Malrotation of midgut in infancy and childhood. Arch Surg 1985; 77: 483-491. 3. Gardner CE, Hart D. Anomalies of intestinal rotation as a cause of intestinal obstruction. Arch Surg 1934; 29: 942-981. 4. Devlin HB. Midgut malrotation causing intestinal obstruction in adult patients. Ann Roy Coll Surg Engl 1971; 48: 227-237. 5 . Cathcart RS, Williamson B, Gregorie HB, Glasow PF. Surgical treatment of midgut, nourotation in the adult patient. Surg Gynecol Obstet 1981; 152: 207-210. 6. Balthazar EJ. Intestinal malrotation in adults. Roentgenographic assessment with emphasis on
9.
10.
11.
12.
13.
Jpn. J. Surg. May 1990 isolated complete and partial nonrotation. 1976; 126: 358-367. RowsonJT, Sullivan SN, Girvan DP. Midgut volvulus in the adult. A complication of intestinal malrotation.J Clin Gastroenterol 1987; 9: 212-216. Van Wingerden GI, Dons RF. Complete duodenal obstruction during pregnancy with intestinal nonrotation and painless midgut volvulus. J Reprod Med 1981; 26: 265-267. Fisher Jig Computed tomogTaphic diagnosis of volvulus in intestinal malrotation. Radiology 1981; 81: 145-146. Ellenberg DJ, Delcastillo J. Duodenal obstruction from peritoneal (Ladd's) bands in a ten-year-old child. Ann Emerg Med 1984; 13: 56-59. Ichimura T, Akashi F, Ishikawa Y, Inoue T, Inoue M. A case of intestinal malrotation with duodenal stenosis and occlusion of the superior mesenteric vein.JapanJ Gastroenterol Surg 1984; 81: 291. Ingram NP, Garnder BP. An unusual presentation of intestinal malrotation in an adult. Brit J Clln Pract 1981; 35: 166-167. Stauffer UG, Herrmann P. Comparison of late results in patients with corrected intestinal realrotation with and without fixation of the mesentery.J Pediatr Surg 1980; 15: 9-12.
duodenal obstruction, acute appendicitis, intestinal mal-
company malrotation. We report herein an INTRODUCTION
D u o d e n a l obstruction secondary to intestinal malrotation is not so rare in neonates and young children but is quite rare in adults. 1-3 T h e related complication seen in adults is obstructions of the small intestine resulting from a midgut volvulus 3-9 and most documentated cases of duodenal obstruction are related to either midgut malrotafionS,4,s, 9 or peritoneal bands.l~ 11 Making an immediate diagnosis of acute abdomen in asymptomatic adult malrotation can be difficult if the physician has limited experience with the various types o f complications that may ac-
*The Department of Surgery, Medical Institute of Bioregutation, Kyushu University,Japan **The Department of Surgery, Oita National Hospital, Oita,Japan ***The Departmentof Gastroenterology, Oita National Hospital, Oita,Japan Reprint requests to: Hiroaki Ueo, MD, The Department of Surgery, Medical Institute of Bioregulation, Kyushu University69, 4546 Tsurumihara, Beppu 874, Japan
unusual case of complete duodenal obstruc-
tion caused by acute appendicitis in a 55 year old man. A CASE REPORT A 55 year old m a n was admitted to our department on January 4, 1988, with complaints of continuous nausea and recent episodes o f severe midabdominal pain and high fever. Eight days prior to his admission, he experienced severe midabdominal pain in the area of the umbilicus. Two days later, a gastrofiberscopic examination showed no abnormal findings in the stomach or duodenal bulbus, after which the abdominal pain diminished spontaneously for 2 days then re-occurred in conjunction with abdominal tenderness and a body temperature of over 39~ He visited his physician again and was prescribed antibiotics for the next 3 days, however, the abdominal pain and fever persisted and he began to complain of nausea and vomiting on the third day of this treatment. T h e patient was then transferred
JAPANESEJOURNALOFSURGERY,VOL.20, No. 3 pp. 346-350, 1990
Volume 20 Number 3
347
Duodenal obstruction caused by appendicitis
Fig. 2. Barium meal study. The duodenal mucosa at the obstruction (see arrow) was normal, suggesting compression by the surrounding tissues.
Fig. 1. Plain X-ray film revealed enlargement of the stomach and duodenum. Since the position of the duodenum appeared to be normal, intestinal malrotation was not diagnosed. to our hospital for detailed investigations. On admission, the patient had no abdominal pain, abdominal distention or fever but had been constipated for 3 days. A nasogastric tube was inserted for gastric and bile juice collection. Vomiting was controlled by fasting and total parenteral nutrition was commenced. Physical examination revealed clinical dehydration and slight distention in the upper region of the umbilicus but no rebound tenderness. A leukocytosis o f ll,200/mm 3 suggested an inflammatory focus. The erythrocyte sedimentation rate was 15/36 mm and c-reative protein was 3.36 u/ml. A plain X-ray film showed dilatation of the stomach and d u o d e n u m (Fig. 1). A barium meal revealed obstruction at the duodeno-jejunal junction seemingly caused by a compression by the surrounding tissues rather than by a duodenal mucosal lesion (Fig. 2). Computed tomography showed no evidence of a low density area in the pancreas or retroperitoneum. A barium e n e m a revealed the caecum to be located in a slightly higher and inner position than nor-
mal and the appendix was not clearly evident (Fig. 3A). Nevertheless, a diagnosis o f intestinal malrotation and acute appendicitis was not made. Since the amount o f gastric and bile juice aspirate increased progressively, a laparotomy was performed on january 25, 1988, which revealed a complete malrotation, and showed the caecum and fight colon to be mobile and high, and advanced appendicitis. An abnormal mesenteric fixation was also present and fibrous adhesion caused by the inflamed appendix involved the duodenojejunal junction at the mesenteric pouch (Fig. 4). Ladd's band was present but was not the cause o f the duodenal obstruction. With appendectomy and release o f the adhesion, the duodenal obstruction was completely reduced (Fig. 3B). T h e patient's postoperative course was uneventful and he has had no further complaints over the subsequent 15 months. Histological examination showed acute inflammatory infiltration in the appendix wall and no malignancy. DiscussioN Most symptomatic anomalies of intestinal r o t a t i o n are detected in the n e o n a t a l period. 1,~ In fact, Kiesewetter and Smith = stated that half the n u m b e r o f such cases are diagnosed during the first postnatal week
Ueo et al.
348
JpnjviJy Surg. 1990
Fig. 3. A Barium enema showing a mobile caecum and poorly identified appendix (arrows). B Postoperative barium meal study.
Fig. 4. IUustration of malrotation and inflamed appendix obstructing the duodenojejunal junction. and eighty per cent within the first month. O n the other hand, most occurrences in
adults are asymptomatic and some are incidentally discovered at routine screenings or during a laparotomy for other disorders. The most frequent symptomatic presentation in adults is intestinal obstruction with midgut volvulus. G a r d n e r and H a r t a reviewed 105 cases of intestinal obstruction caused by abnormalities o f intestinal rotation and stated that 88 were caused by a volvulus of the entire mesentery, 10 by obstruction of the transverse colon and 7 by obstruction o f the d u o d e n u m by a b n o r m a l intestinal fixation. Delvin 4 reported 7 adult cases o f midgut malrotation causing intestinal obstruction and Cathcart et al. ~ reported 5 adult patients with midgut nonrotation. Balthazar 6 assessed roentgenographic presentations in 28 adult cases and noted two different patterns, one b e i n g the classical f o r m of nonrotation of the entire midgut and the other, the isolated form o f complete a n d partial rotation. In most of these reported cases of midgut malrotation in adults, however, abdominal disorders h a d b e e n p r e s e n t since childh o o d ? -~ Two cases of midgut volvulus first
Volume 20 Number 3
Duodenal obstruction caused by appendicitis
349
TaMe 1. Cases of Duodenal Obstruction with Anomalies of Intestinal Rotation Reporters
Total Cases
Duodenal Obstruction
Midgut malrotation
Gardner and Hart s Delvin4 Van Wingerden and Dons 8 FisherJ.K. 9
46 7 1 1
6 2 1 1
Abnormal intestinal fixation
Gardner and Hart s
2
2
Peritoneal bands (Ladd's band)
Ellenberg and Delcastillo 1~ Ichimura T. et alY
1" 1
1" 1
Acute appendicitis
Ueo et al. (the present case)
1
1
Cause
*; a 10 year old child; all others were over the age of 14 years.
occurring in middle age were reported by Rowson et al., 7 and our patient h a d also b e e n symptom-free until the acute onset o f appendicitis caused the duodenal obstruction. The documented cases of duodenal obstruction occurring in adults and adolescents with anomalies of intestinal rotation are summarized in Table 1. Duodenal obstruction is usually related to midgut malrotation Or p e r i t o n e a l b a n d s . I n o n e series 3 o f anomalies o f intestinal rotation, 17 o f 88 patients with midgut malrotation h a d a duodenal obstruction. Moreover, in the same report, there were 7 cases o f duodenal obstruction caused by a b n o r m a l intestinal fixation, 2 of w h o m were adults. Van Wingerden and Dons 8 reported complete duodenal obstruction in a 24 year old p r e g n a n t w o m a n with intestinal malrotation and midgut volvulus. Ellenberg a n d Delcastillo 1~ reported a case of duodenal obstruction from peritoneal (Ladd's) bands and I c h i m u r a et alY treated an adolescent with duodenal stenosis caused by Ladd's bands and intestinal malrotation. However, our study of the literature revealed no documentation o f duodenal obstruction caused by acute appendicitis with intestinal malrotation. As for appendicitis causing intestinal obstruction in the case o f malrotation in adults, I n g r a m and Garnder 12 reported one case o f a n obstruction o f the ileum in the left iliac fossa, in the presence of
an inflamed appendix. B e c a u s e the caecum is often located in the u p p e r portion around the d u o d e n u m in patients with malrotation and because acute appendicitis is c o m m o n in adults, duodenal obstruction from acute appendicitis may occur more frequently than expected. O n e pertinent clinical indication of duodenal obstruction is a copious a m o u n t of bile-stained vomitus. Plain X-ray film and barium meal study clearly revealed a dilated duodenal loop and the findings o f the barium e n e m a supported the diagnosis of intestinal malrotation with a mobile caecum. A poorly identified appendix on the barium e n e m a in c o n j u n c t i o n with t e n d e r n e s s around the mobile caecum also suggested an inflamed appendix. W h e n patients with vomiting caused by duodenal obstruction have tenderness in the u p p e r region o f the umbilicus, the possibility of acute appendicitis with intestinal malrotation should be considered, especially in patients with previous episodes o f gastrointestinal complaints. T h e surgical treatment involves releasing the adhesion which obstructs the d u o d e n u m and removing the appendix. T h e intestine is then returned to its normal position and no particular fixation is required. Stauffer and H a r r m a n n 13 c o m p a r e d the late results of 77 surgical patients with intestinal malrotation who underwent or did not undergo additional intestinal fixation. T h e y found that
350
Ueo et al.
intestinal fixation reduced neither the incidence of reoperation nor that of postoperative abdominal complaints. (Received for publication onJun. 19, 1989)
7.
8.
REFERENCES 1. Stewart DR, Colondy AL, Daggett WC. Malrotation of the bowel in infants and children: A 15 year review. Surg 1976; 79: 716-720. 2. Kiesewetter WB, Smith JW. Malrotation of midgut in infancy and childhood. Arch Surg 1985; 77: 483-491. 3. Gardner CE, Hart D. Anomalies of intestinal rotation as a cause of intestinal obstruction. Arch Surg 1934; 29: 942-981. 4. Devlin HB. Midgut malrotation causing intestinal obstruction in adult patients. Ann Roy Coll Surg Engl 1971; 48: 227-237. 5 . Cathcart RS, Williamson B, Gregorie HB, Glasow PF. Surgical treatment of midgut, nourotation in the adult patient. Surg Gynecol Obstet 1981; 152: 207-210. 6. Balthazar EJ. Intestinal malrotation in adults. Roentgenographic assessment with emphasis on
9.
10.
11.
12.
13.
Jpn. J. Surg. May 1990 isolated complete and partial nonrotation. 1976; 126: 358-367. RowsonJT, Sullivan SN, Girvan DP. Midgut volvulus in the adult. A complication of intestinal malrotation.J Clin Gastroenterol 1987; 9: 212-216. Van Wingerden GI, Dons RF. Complete duodenal obstruction during pregnancy with intestinal nonrotation and painless midgut volvulus. J Reprod Med 1981; 26: 265-267. Fisher Jig Computed tomogTaphic diagnosis of volvulus in intestinal malrotation. Radiology 1981; 81: 145-146. Ellenberg DJ, Delcastillo J. Duodenal obstruction from peritoneal (Ladd's) bands in a ten-year-old child. Ann Emerg Med 1984; 13: 56-59. Ichimura T, Akashi F, Ishikawa Y, Inoue T, Inoue M. A case of intestinal malrotation with duodenal stenosis and occlusion of the superior mesenteric vein.JapanJ Gastroenterol Surg 1984; 81: 291. Ingram NP, Garnder BP. An unusual presentation of intestinal malrotation in an adult. Brit J Clln Pract 1981; 35: 166-167. Stauffer UG, Herrmann P. Comparison of late results in patients with corrected intestinal realrotation with and without fixation of the mesentery.J Pediatr Surg 1980; 15: 9-12.
