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Interval laparoscopic appendectomy for appendicitis complicated by pylephlebitis.
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MedLine Citation:
PMID:  16709373     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: Although rare, portal mesenteric venous thrombosis and pylephlebitis remain potential life-threatening sequelae of ruptured appendicitis in children. Treatment recommendations from recent reports have included urgent exploratory laparotomy with appendectomy, prolonged intravenous antibiotic therapy, and anticoagulation for up to a year.
METHODS: This report describes successful management of pylephlebitis and mesenteric venous thrombosis complicating ruptured appendicitis with intravenous antibiotics and anticoagulation followed by interval laparoscopic appendectomy.
RESULTS: A previously healthy 5-year-old girl was diagnosed with ruptured appendicitis complicated by pylephlebitis and mesenteric venous thrombosis at the time of presentation. She was treated with intravenous antibiotics and anticoagulated for 3 months. She subsequently underwent interval laparoscopic appendectomy. At 3-year follow-up, she is healthy without evidence of adverse sequelae.
DISCUSSION: This is the first reported case of successful, minimally invasive management of ruptured appendicitis complicated by mesenteric venous thrombosis and pylephlebitis.
CONCLUSION: Similar treatment of other children with this rare presentation seems reasonable.
Authors:
Karyn B Stitzenberg; Mark D Piehl; Paul E Monahan; J Duncan Phillips
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Publication Detail:
Type:  Journal Article; Review    
Journal Detail:
Title:  JSLS : Journal of the Society of Laparoendoscopic Surgeons / Society of Laparoendoscopic Surgeons     Volume:  10     ISSN:  1086-8089     ISO Abbreviation:  JSLS     Publication Date:    2006 Jan-Mar
Date Detail:
Created Date:  2006-05-19     Completed Date:  2006-09-26     Revised Date:  2013-03-26    
Medline Journal Info:
Nlm Unique ID:  100884618     Medline TA:  JSLS     Country:  United States    
Other Details:
Languages:  eng     Pagination:  108-13     Citation Subset:  IM    
Affiliation:
Cecil G. Sheps Center for Health Services Research and Department of Surgery, University of North Carolina, Chapel Hill, North Carolina, USA.
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MeSH Terms
Descriptor/Qualifier:
Anti-Bacterial Agents / therapeutic use
Anticoagulants / therapeutic use
Appendectomy / methods*
Appendicitis / surgery*
Child, Preschool
Female
Humans
Laparoscopy*
Mesenteric Vascular Occlusion / complications,  drug therapy
Mesenteric Veins
Portal Vein*
Thrombophlebitis / complications*,  drug therapy
Venous Thrombosis / complications,  drug therapy
Chemical
Reg. No./Substance:
0/Anti-Bacterial Agents; 0/Anticoagulants
Comments/Corrections

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

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Journal Information
Journal ID (nlm-ta): JSLS
Journal ID (hwp): jsls
Journal ID (pmc): jsls
Journal ID (publisher-id): JSLS
ISSN: 1086-8089
ISSN: 1938-3797
Publisher: Society of Laparoendoscopic Surgeons, Miami, FL
Article Information
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© 2006 by JSLS, Journal of the Society of Laparoendoscopic Surgeons.
open-access:
Print publication date: Season: Jan–Mar Year: 2006
Volume: 10 Issue: 1
First Page: 108 Last Page: 113
ID: 3015684
PubMed Id: 16709373

Interval Laparoscopic Appendectomy for Appendicitis Complicated by Pylephlebitis
Karyn B. Stitzenberg, MD, MPH Affiliation: Cecil G. Sheps Center for Health Services Research and Department of Surgery, University of North Carolina, Chapel Hill, North Carolina, USA.
Mark D. Piehl, MD Affiliation: Department of Pediatrics, University of North Carolina, Chapel Hill, North Carolina and WakeMed Hospital, Raleigh, North Carolina, USA.
Paul E. Monahan, MD Affiliation: Department of Pediatrics, University of North Carolina, Chapel Hill, North Carolina, USA.
J. Duncan Phillips, MD Affiliation: Department of Surgery, University of North Carolina, Chapel Hill, North Carolina and WakeMed Hospital, Raleigh, North Carolina, USA.
Financial Disclosure: None of the authors have any financial interest in any commercial device, equipment, instrument, or drug pertinent to this case report. No financial support was received for this report. None of the authors have any conflict of interest pertinent to this report.
[presented-at] Presented as an abstract at the annual meeting of the International Pediatric Endosurgery Group, Maui, Hawaii, USA May 2004.
Correspondence: Address reprint requests to: J. Duncan Phillips, MD, Department of Surgery, Division of Pediatric Surgery, 3010 Old Clinic Bldg, CB#7223, Chapel Hill, NC 27599-7223, USA. Telephone: 919 966 4220, Fax: 919 843 2497

INTRODUCTION

Mesenteric venous thrombosis and pylephlebitis are rare sequelae of ruptured appendicitis in children. Traditional management has included urgent exploratory laparotomy with appendectomy, intravenous antibiotics, and long-term anticoagulation. A recent review of the English-language literature revealed no published reports of management of appendicitis complicated by pylephlebitis with minimally invasive surgical techniques. We describe successful management with intravenous antibiotics and anticoagulation followed by interval laparoscopic appendectomy in a 5-year-old girl.


CASE REPORT

A previously healthy 5-year-old girl presented with a 7-day history of fever, vomiting, and anorexia and a 24-hour history of diffuse abdominal pain. At the time of presentation, her rectal temperature was 104.1°F. Physical examination demonstrated generalized voluntary abdominal guarding but no focal tenderness. Notable laboratory abnormalities included white blood count 14.5 (x109/L) with 83% polymorphonuclear leukocytes, aspartate amino transferase (AST) 66 U/L, amino alanine transferase (ALT) 107 U/L, alkaline phosphatase 267 U/L, and total bilirubin 1.1 mg/dL. Computed tomography (CT) of the abdomen and pelvis was obtained and revealed a thickened retrocecal appendix with associated inflammatory changes but no focal fluid collection (Figure 1), thrombosis of the superior mesenteric vein (Figure 2) with extension of the thrombus into the left branch of the portal vein, and impaired perfusion of the left lobe of the liver (Figure 3).

The patient was admitted to the hospital, hydrated, placed on broad-spectrum intravenous (IV) antibiotics, and anticoagulated with IV unfractionated heparin. Her fevers, nausea, and pain quickly resolved. A diet was offered on hospital day 4, which she tolerated without difficulty. On hospital day 6, a follow-up CT scan was obtained and demonstrated dramatic improvement in hepatic perfusion (Figure 4), despite residual superior mesenteric vein thrombus. After 12 days of IV antibiotics and heparin, she was discharged home. Discharge medications included low-molecular weight heparin, enoxaparin 40 mg sq qd (Lovenox, Aventis Pharmaceuticals, Inc, France). She was subsequently converted to oral warfarin, dose-adjusted to maintain an international normalized ratio (INR) between 2.0 and 3.0.

Pediatric hematology consultation was obtained, and a hypercoagulability workup was initially consistent with heterozygous protein C deficiency: activity 35% (normal, 64% to 126%). Antithrombin III level was also mildly low at 60% (normal, 70% to 115%). Protein C antigen level while the patient was on heparin was 99% (normal, 71% to 157%). However, coagulation functional screens obtained at the time of clot presentation are known to be unreliable. After 3 months, the warfarin was discontinued; repeat testing at that time revealed no evidence of protein C deficiency (activity 82% of normal) nor of antithrombin III deficiency (101% of normal).

Four months after initial presentation, the patient underwent laparoscopic appendectomy. Intraperitoneal examination revealed mild scarring and evidence of previous inflammation, but no other abnormality (Figure 5). The appendix was removed without difficulty by using a 3-trocar technique. Operative time was 68 minutes. Subsequent histologic examination of the appendix revealed fibrosis and evidence of prior sealed perforation. She had an uncomplicated postoperative course and was discharged home on the third postoperative day. She is now 3 years postappendectomy with no evidence of long-term sequelae. Notably, she has had no further thrombotic events.


DISCUSSION

Before the second half of the 20th century, the incidence of mesenteric venous thrombosis and pylephlebitis complicating appendicitis was less than 1%.1 Since the advent of modern antibiotic therapy and aggressive surgical approaches to appendicitis, pylephlebitis has become an exceedingly rare, yet still potentially fatal, complication of appendicitis. Although the low incidence makes it difficult to know the true mortality rate of pylephlebitis secondary to appendicitis, modern estimates of the mortality of pylephlebitis from all causes are as high as 50%.2,3 Although pylephlebitis may rarely occur shortly after the onset of symptoms, it most often is associated with cases of appendicitis in which a delay occurs in presentation or diagnosis. Alternatively, pylephlebitis occasionally occurs after appendectomy in cases of ruptured appendicitis. Because the presentation of appendicitis can be atypical in young children, they are at increased risk for delayed presentation to a health care provider and diagnosis. Consequently, children may be particularly vulnerable to pylephlebitis.

Traditional management of appendicitis complicated by pylephlebitis has included intravenous antibiotic therapy and emergent exploratory laparotomy with appendectomy. The need for long-term anticoagulation has been a topic of debate for the last century and remains disputed.2, , 6 A review of the English-language literature by using PubMed (National Center for Biotechnology Information, Bethesda, MD) yielded 24 articles published since 1959, describing 34 cases of mesenteric venous thrombosis and pylephlebitis complicating appendicitis. Additionally, a review by Klinefelter et al7 details 62 cases reported between 1926 and 1959. Of the 34 reported cases since 1959, only 4 occurred in females. This is consistent with Klinefelter's observation that roughly 85% of cases of pylephlebitis secondary to appendicitis occur in males.7 Forty-four percent (15/34) of the cases occurred in children (Table 1). In the children, pylephlebitis was present at the time of diagnosis of appendicitis in 10 cases. Seven of these patients were treated with urgent exploratory laparotomy and appendectomy, and several of these patients went on to require additional procedures. Two of the 10 patients were treated with interval open procedures, and our current patient was treated with an interval laparoscopic appendectomy. For the remaining 2 patients, no description of the surgical management was included in the report. Of the 3 patients in whom onset of pylephlebitis occurred after appendectomy, 1 patient required subsequent second and third laparotomies, while 2 were managed nonoperatively.

To our knowledge, the case described in this report is the first case of appendicitis complicated by pylephlebitis that was treated using a minimally invasive approach. Using anticoagulation and intravenous antibiotics, we were able to successfully manage the acute presentation nonoperatively. Consequently, we were able to subsequently perform an interval appendectomy laparoscopically. This approach averted the morbidity associated with emergent open procedures.


CONCLUSION

This is the first reported case of minimally invasive management of appendicitis complicated by pylephlebitis and mesenteric venous thrombosis. Based on our experience we feel that, for children with this rare presentation, interval laparoscopic appendectomy, after a course of anticoagulation and intravenous antibiotics, is an acceptable alternative to routine emergent exploratory laparotomy and appendectomy.


References:
1.. Soro Y. Pylephlebitis and liver abscesses due to appendicitis. J Int Coll Surg. Year: 1948; XI: 464–46818885819
2.. Plemmons RM,Dooley DP,Longfield RN. Septic thrombophlebitis of the portal vein (pylephlebitis): diagnosis and management in the modern era. Clin Infect Dis. Year: 1995; 21: 1114–11208589130
3.. Baril N,Wren S,Radin R,Ralls P,Stain S. The role of anticoagulation in pylephlebitis. Am J Surg. Year: 1996; 172: 449–452; discussion 452–453. 8942542
4.. Clark CW Jr.,Bunn PA. Treatment of pylephlebitis secondary to acute appendicitis. N Y State J Med. Year: 1953; 53: 3007–301013111508
5.. Balthazar EJ,Gollapudi P. Septic thrombophlebitis of the mesenteric and portal veins: CT imaging. J Comput Assist Tomogr. Year: 2000; 24: 755–76011045699
6.. Duffy FJ Jr.,Millan MT,Schoetz DJ Jr.,Larsen CR. Suppurative pylephlebitis and pylethrombosis: the role of anticoagulation. Am Surg. Year: 1995; 61: 1041–10447486441
7.. Klinefelter HF Jr.,Grose WE,Crawford HJ. Pylephlebitis Bull Johns Hopkins Hosp. Year: 1960; 106: 65–73
8.. Babcock DS. Ultrasound diagnosis of portal vein thrombosis as a complication of appendicitis. AJR Am J Roentgenol. Year: 1979; 133: 317–319110102
9.. Shaw PJ,Saunders AJ,Drake DP. Case report: ultrasono-graphic demonstration of portal vein thrombosis in the acute abdomen. Clin Radiol. Year: 1986; 37: 101–1023514073
10.. Giuliano CT,Zerykier A,Haller JO,Wood BP. Radiological case of the month. Pylephlebitis secondary to unsuspected appendiceal rupture. Am J Dis Child. Year: 1989; 143: 1099–11002773888
11.. Slovis TL,Haller JO,Cohen HL,Berdon WE,Watts FB Jr. Complicated appendiceal inflammatory disease in children: pylephlebitis and liver abscess. Radiology. Year: 1989; 171: 823–8252655006
12.. Scully RE ed. Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises. Case 22-1991. A 15-year-old boy with fever of unknown origin, severe anemia, and portal-vein thrombosis. N Engl J Med. Year: 1991; 324 (22): 1575–15842027361
13.. van Spronsen FJ,de Langen ZJ,van Elburg RM,Kimpen JL. Appendicitis in an eleven-year-old boy complicated by thrombosis of the portal and superior mesenteric veins. Pediatr Infect Dis J. Year: 1996; 15: 910–9128895927
14.. Eire PF,Vallejo D,Sastre JL,Rodriguez MA,Garrido M. Mesenteric venous thrombosis after appendicectomy in a child: clinical case and review of the literature. J Pediatr Surg. Year: 1998; 33: 1820–18219869061
15.. Kader HA,Baldassano RN,Harty MP,et al. Ruptured retrocecal appendicitis in an adolescent presenting as portal-mesenteric thrombosis and pylephlebitis. J Pediatr Gastroenterol Nutr. Year: 1998; 27: 584–5889822327
16.. Schmutz GR,Benko A,Billiard JS,Fournier L,Peron JM,Fisch-Ponsot C. Computed tomography of superior mesenteric vein thrombosis following appendectomy. Abdom Imaging. Year: 1998; 23: 563–5679922185
17.. Vanamo K,Kiekara O. Pylephlebitis after appendicitis in a child. J Pediatr Surg. Year: 2001; 36: 1574–157611584411
18.. Chang TN,Tang L,Keller K,Harrison MR,Farmer DL,Albanese CT. Pylephlebitis, portal-mesenteric thrombosis, and multiple liver abscesses owing to perforated appendicitis. J Pediatr Surg. Year: 2001; 36: E1911528636
19.. Nanni L,Vallasciani S,D'Urzo C,et al. Bilateral renal vein thrombosis as a complication of gangrenous appendicitis. Pediatr Med Chir. Year: 2002; 24: 237–23912236042
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Figures

[Figure ID: F1]
Figure 1. 

Thickened appendix with associated inflammatory changes.



[Figure ID: F2]
Figure 2. 

Thrombosis of the superior mesenteric vein.



[Figure ID: F3]
Figure 3. 

Impaired perfusion of the left lobe of the liver, secondary to extension of thrombus into the left branch of the main portal vein.



[Figure ID: F4]
Figure 4. 

Improved perfusion of the liver after 6 days of intravenous antibiotics and anticoagulation.



[Figure ID: F5]
Figure 5. 

Mild scarring and postinflammatory changes in the right lower quadrant at the time of interval laparoscopic appendectomy 4 months following acute appendicitis, pylephlebitis, and mesenteric venous thrombosis.



Tables
[TableWrap ID: T1] Table 1. 

Details of the 15 Reported Cases of Mesenteric Venous Thrombosis and Pylephlebitis Complicating Appendicitis in Children Since 1959


Author Year Age Sex Operative Management Anticoagulation
Babcock8 1979 6 M Exploratory laparotomy with appendectomy, second laparotomy with liver abscess drainage No
Shaw9 1986 13 M Exploratory laparotomy with appendectomy and drainage of periappendiceal abscess No
Giuliano10 1989 12 M Exploratory laparotomy with appendectomy No
Slovis11 1989 6 F Not stated Not stated
Slovis11 1989 9 M Not stated Not stated
Scully (Ed.)12 1991 15 M Exploratory laparotomy with ileocectomy and liver biopsy No
van Spronsen13 1996 11 M Interval open appendectomy No
Eire14 1998 12 F Exploratory laparotomy, second-look exploratory laparotomy (occurred subsequent to open appendectomy) Yes
Kader15 1998 15 M Interval exploratory laparotomy with appendectomy Yes
Schmutz16 1998 18 M No additional surgery performed (occurred subsequent to appendectomy) No
Vanamo17 2001 7 F Exploratory laparotomy with appendectomy and drainage periappendiceal abscess Yes
Chang18 2001 8 M Exploratory laparotomy with ileocectomy and drainage of liver abscesses Yes
Nanni19 2002 10 M No additional surgery performed (occurred subsequent to appendectomy) No
Pitcher20 2003 17 M Exploratory laparotomy with appendectomy Yes
Stitzenberg 2006 5 F Interval laparoscopic appendectomy Yes


Article Categories:
  • Case Reports

Keywords: Pylephlebitis, Interval appendectomy, Laparoscopic appendectomy, Acute appendicitis.

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