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Acute appendicitis complicated by necrotizing fasciitis in a teenager
Journal of Pediatric Surgery Case Reports 37 (2018) 77–82
Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc
Acute appendicitis complicated by necrotizing fasciitis in a teenager a
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Jeremy D. Kauffman , Megan O'Brien , Christopher W. Snyder , S. Alex Rottgers , Drew A. Rideouta, Nicole M. Chandlera,∗ a b
Division of Pediatric Surgery, Johns Hopkins All Children's Hospital, 601 5th Street South, Suite 306, St. Petersburg, Florida 33701, USA Division of Plastic and Reconstructive Surgery, Johns Hopkins All Children's Hospital, 601 5th Street South, Suite 306, St. Petersburg, Florida 33701, USA
A R T I C LE I N FO
A B S T R A C T
Keywords: Necrotizing fasciitis Appendicitis Pediatrics Abdominal wall reconstruction Synthetic marijuana
Necrotizing fasciitis is a rare complication of appendicitis in children and is associated with significant morbidity and mortality. We present the case of a 16-year-old male who presented with perforated appendicitis and subsequently developed necrotizing fasciitis of the abdominal wall and perineum. His seven-week hospital course was notable for 16 trips to the operating room, which culminated in staged, complex abdominal wall repair. Ultimately he was discharged home in good condition. The case is remarkable for the extent of soft tissue damage and complexity of repair precipitated by an uncommon complication of a common pediatric disease.
1. Introduction Appendicitis is a common cause of an acute abdomen in children [1]. Delay in treatment increases the likelihood of complications [2]. Necrotizing fasciitis is an uncommon complication of appendicitis, particularly in children, but is associated with significant morbidity and mortality [3]. We present the case of a 16-year-old male who presented with perforated appendicitis, underwent laparoscopic appendectomy, and developed necrotizing fasciitis. The case illustrates the need for timely diagnosis and aggressive surgical management of necrotizing fasciitis, as well as the need for vigilance in working-up even the most routine of pediatric diseases. 2. Case report A 16 year-old, previously healthy male presented to a community hospital with abdominal pain, scrotal swelling, and signs of dehydration and sepsis. The abdominal pain had been present for three days but the patient had delayed seeking medical care because he attributed the pain to withdrawal from synthetic (“spice”) marijuana. He also reported having been struck in the groin during wrestling practice six days prior to presentation. Testicular ultrasound demonstrated a large hydrocele and what appeared to be a bowel-containing right inguinal hernia. Computed tomography (CT) showed evidence of inflammation in the right lower quadrant and extraluminal air extending into the right scrotum (Fig. 1). The differential diagnosis at this time included perforated appendicitis and traumatic perforation of herniated bowel.
Fluid resuscitation and empiric antimicrobial therapy with piperacillintazobactam were initiated. The patient was transferred to a regional hospital where he was taken to the operating room (OR) for diagnostic laparoscopy, which revealed perforated appendicitis and diffuse peritonitis with no evidence of inguinal hernia. Laparoscopic appendectomy with abdominal washout was performed. The scrotum was aspirated percutaneously due to concern for local extension of the infection, but no purulent material was aspirated and no further intervention was performed. The patient's condition initially improved, but he exhibited persistent tachycardia and developed erythema and bullous changes of the right scrotum and inguinal region, for which clindamycin was added to his antibiotic regimen. Multiple sets of blood cultures drawn at this time grew no organisms. He was taken back to the OR on postoperative day five, at which time he was found to have necrotizing fasciitis of the perineum and right groin. Local debridement was performed and a negativepressure wound vacuum applied. Postoperatively, he remained intubated and required ionotropic support. Gram stain of scrotal and abdominal fluid demonstrated gram negative rods and gram positive cocci. Cultures eventually grew Escherichia coli with intermediate resistance to piperacillin-tazobactam, as well as Bacteroides fragilis and Streptococcus constellatus. Meropenem and vancomycin were initiated, and piperacillin-tazobactam was discontinued. Two days later he remained in critical condition and was transferred to a quaternary care pediatric hospital. His APACHE IV score at the time of transfer was 70, with a corresponding mortality risk of 37.3% [4]. He was taken to the OR for debridement of the scrotum, abdominal wall,
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Corresponding author. 601 5th St. South, Dept 70-6600, 3rd Floor, St. Petersburg, Florida, 33701 USA. E-mail addresses: jkauff[email protected] (J.D. Kauffman), [email protected] (M. O'Brien), [email protected] (C.W. Snyder), [email protected] (S.A. Rottgers), [email protected] (D.A. Rideout), [email protected] (N.M. Chandler). https://doi.org/10.1016/j.epsc.2018.07.018 Received 17 July 2018; Accepted 21 July 2018 Available online 25 July 2018 2213-5766/ © 2018 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
Journal of Pediatric Surgery Case Reports 37 (2018) 77–82
J.D. Kauffman et al.
Fig. 3. Appearance of wound on hospital day seven following initial four debridements. Surgical concerns at this time included: 1) full thickness abdominal all defect with exposed liver (arrow) and colon (arrowhead) 2) exposed bilateral testicles (circles) and 3) massive skin defect.
complex abdominal wall reconstruction. Control of the infection ultimately required removal of the external oblique, serratus, and inferior latissimus dorsi muscles on the right side, resulting in a right flank wound measuring 68 × 15 cm, right leg wound measuring 30 × 8 cm, and a left flank and perineum wound measuring 30 × 8 cm. The full thickness defect resulted in exposure of the liver, colon, and bilateral testicles (Fig. 3). Reconstruction of the right abdominal wall was achieved in a staged fashion by means of an acellular dermal matrix (Fig. 4), gracilis flap, and vastus lateralis flap (Fig. 5). Repair on the left side was accomplished with primary closure over a drain (Fig. 6). The patient's course was complicated early on by pneumonia, which
Fig. 1. CT at time of initial presentation. Coronal view demonstrating 1) appendicolith (arrow), 2) extraluminal air in the peritoneum and retroperitoneum (arrowheads), and 3) pneumoscrotum (circle).
perineum, right lateral chest wall and flank (Fig. 2). Over the course of the next three weeks he was taken to the OR an additional ten times for wound debridement, negative pressure dressing application, and
Fig. 2. Before (panel A) and after (panels B–D) initial debridement at the children's hospital.
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Fig. 4. Initial closure of right-sided, full-thickness abdominal wall defect with an acellular dermal matrix.
Fig. 6. Partial primary closure of the left perirectus wound over a drain.
Fig. 7. Right flank wound covered with cadaveric skin graft.
Metronidazole was eventually added for better anaerobic coverage, and fluconazole was added when Candida albicans grew from abdominal wound and pleural fluid cultures. Linezolid was added late in his course for growth of Staphylococcus haemolyticus from the left groin wound. Antibiotic and antifungal therapy were ultimately discontinued seven weeks after his initial presentation. Temporary skin closure with cadaver graft was performed (Fig. 7) in order to allow time for nutritional optimization and wound granulation prior to definitive closure with split-thickness skin grafting. Nine weeks and five days after his initial presentation, he was discharged home. When he returned to clinic for follow-up four weeks later, his wounds were healing well with less than one percent total surface area of breakdown throughout the skin graft (Fig. 8). With the exception of a brief admission for drainage of an abscess of the medial right thigh, the patient has continued to progress well and has returned to his normal activities of daily living. Fig. 5. Reconstruction of the (A) right abdominal wall with acellular dermal matrix and gracilis flap and (B) vastus lateralis muscle flap mobilized and reflected proximally to cover right flank mesh repair.
3. Discussion The case described above is notable on several accounts. First, necrotizing fasciitis is an uncommon complication of appendicitis, particularly in the pediatric population [5,6]. In our review of the literature, which was not restricted by language and included searches in PubMed, Ovid MEDLINE, and Embase, we identified a total of 42 cases (Appendix 1). Median age of the cohort was 65 years (IQR = 44–77).
improved following treatment with parenteral antibiotics and antifungals. Later in his course he developed an enterocutaneous fistula, which resolved with conservative therapy. Meropenem was continued throughout his course, while vancomycin was discontinued shortly after his third operation with concern for worsening acute renal failure.
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Fig. 8. Wound appearance at one month follow-up.
and even a diagnosis as routine as appendicitis must be considered judiciously.
Only two cases occurred in children. One of these occurred in a 19-dayold male who developed necrotizing fasciitis following appendectomy and subsequently died of multiple organ dysfunction [7]. The second occurred in a ten-year-old male who required repair with a skin graft and survived [8]. When considering patients of all ages identified in our review, in-hospital mortality was 35.1%, and median hospital length of stay was six weeks (IQR = 2–11) among those who survived. The case is also notable for the extent of tissue loss and associated complexity of reconstructive repair. While several reports of necrotizing fasciitis following appendicitis describe repair of the abdominal wall with skin grafts, one abstract mentions the use of regional advancement flaps, and one article describes disarticulation of the hip, we encountered no report of reconstruction with gracilis and vastus lateralis muscle flaps [9–13]. The case demonstrates that excellent functional outcomes can be achieved in the setting of full-thickness loss of portions of the abdominal and perineal wall using a combination of local wound care and staged reconstruction with biologic and autologous grafts. Perhaps of only incidental consequence is the temporal association of synthetic (“spice”) marijuana use and the onset of the patient's initial symptoms. It may be worth noting that synthetic marijuana has been associated with a plethora of untoward side effects, ranging from mild to potentially lethal in severity [14,15]. These include such “bad actors” as seizure, stroke, myocardial infarction, and cardiogenic shock, in addition to the “usual suspects” of vomiting, confusion, and psychosis [16–19]. To our knowledge there have been no reports of appendicitis or necrotizing fasciitis in association with synthetic marijuana. While there is insufficient evidence to support a causal relationship between spice use and our patient's pathology, the patient's perception that his initial symptoms were due to spice withdrawal contributed to a delay in treatment, which in turn resulted in greater morbidity.
Author contribution
• Jeremy Kauffman: Study design, drafting and critical revision of the manuscript, statistical analysis, literature review. • Megan O'Brien: Study design, drafting and critical revision of the manuscript, literature review. • Christopher W. Snyder: Study design, photography, critical revision of the manuscript • S. Alex Rottgers: Study design, photography, critical revision of the manuscript • Drew A. Rideout: Data interpretation, critical revision of the manuscript • Nicole Chandler: Study design, critical revision of the manuscript. Institutional review board The Johns Hopkins Medicine IRB does not require IRB approval for case reports. Therefore IRB approval to publish this care report was not obtained. Patient consent Witten consent of the patient's mother and written assent of the patient to publish this case report were obtained. Funding No funding or grant support.
4. Conclusion
Authorship
In conclusion, this case highlights the fact that necrotizing infection can lead to rapid and extensive soft tissue loss with potentially lifethreatening consequences in children with appendicitis. Timely detection and expeditious triage to an appropriate level of care are critical to survival in these circumstances. Early and aggressive debridement of necrotic tissue, rapid initiation of broad-spectrum antibiotics, and assiduous supportive care with appropriate respiratory and hemodynamic support are the mainstay of treatment [20]. This case demonstrates that children are not immune to the lethal risks posed by this complication,
All authors attest that they meet the current ICMJE criteria for Authorship. Conflict of interest The following authors have no financial disclosures: JDK, MO, CWS, SAR, DAR, NMC.
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Appendix 1. Cases of Appendicitis complicated by necrotizing fasciitis reported in the literature.
No.
Author
Year
Sex
Age∗
In-Hospital Mortality
Length of Stay∗∗
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31–33 34 35 36 37 38 39 40 41 42
Awe[1] Azab[2] Bobrow[3] Cano[4] Chen[5] Coulier[6] Fernandes[7] Fujiwara[8] Garcia-Santos[9] Georgiou[10] Gerber[11] Groth[12] Guirguis[13] Hartley[14] Harwant[15] Hensley[16] Hua[17] Huang[18] Hung[19] Hung[19] Jacobs[20] Ketwong[21] Kjossev[22] Lodha[23] Mai[24] Marinis[25] Marron[26] Mazza[27] Mukoyama[28] Nazir[29] Oztas[30] Penninga[31] Raghavendra[32] Rajaguru[33] Romanoff[34] Şenkul[35] Taif[36] Takeda[35] Tsimogianni[37] Wilharm[38]
2003 2013 1996 2016 2010 2012 2011 1999 2012 2013 1994 1999 1989 1994 2001 2011 2015 2018 2015 2015 1993 2017 2003 2003 2010 2015 2005 1987 2003 2012 2013 2004 2010 2016 2016 2003 2014 2012 2012 2010
Male Male Male Female Female Male Male Female Male Male Male Female Female Female Female Female Male Male Male Female Female Female Female Male Male Female Female Female Male Female NR Female Male Male Male NR Female Male Male Male
28 23 63 62 76 41 10 98 80 84 85 49 80 79 66 69 50 65 73 91 44 63 83 0.05 65 83 67 59 77 72 NR 33 19 47 71 NR 26 76 52 21
No No Yes No No No No No NR No No No NR Yes Yes No Yes Yes No Yes No No Yes Yes No Yes No Yes No No NR No No No No Yes Yes Yes No No
33 60 NA 45 73 NR NR 16 NR 22 NR NR NR NA NA 28 NA NA 60 NA 40 120 NA NA 14 NA NR NA 80 NR NR NR NR NR NR NA NA NA NR 91
NR - Not reported; NA - Not applicable. * Age in years. ** Length of stay in days.
References cited in text [10] [1] Glass CC, Rangel SJ. Overview and diagnosis of acute appendicitis in children. Semin Pediatr Surg 2016 Aug;25(4):198–203. [2] Rentea RM, Peter SDS, Snyder CL. Pediatric appendicitis: state of the art review. Pediatr Surg Int 2017 Mar;33(3):269–83. [3] Romanoff A, Freed J, Heimann T. A case report of necrotizing fasciitis of the abdominal wall: a rare, life-threatening complication of a common disease process. Int J Surg Case Rep 2016;28:355–6. [4] Zimmerman JE, Kramer AA, McNair DS, Malila FM. Acute physiology and chronic health evaluation (Apache) IV: hospital mortality assessment for today's critically ill patients. Crit Care Med 2006 May;34(5):1297–310. [5] Takeda M, Higashi Y, Shoji T, Hiraide T, Maruo H. Necrotizing fasciitis caused by a primary appendicocutaneous fistula. Surg Today 2012 Aug;42(8):781–4. [6] Chen CW, Hsiao CW, Wu CC, Jao SW, Lee TY, Kang JC. Necrotizing fasciitis due to acute perforated appendicitis: case report. J Emerg Med 2010 Aug;39(2):178–80. [7] Lodha A, Wales PW, James A, Smith CR, Langer JC. Acute appendicitis with fulminant necrotizing fasciitis in a neonate. J Pediatr Surg 2003 Nov;38(11):5. [8] Fernandes C, Damaso C, Duarte R, Cardoso DS, Casella P. Necrotizing fasciitis postacute appendicitis. Acta Med Port 2011 Dec;24(Suppl 3):621–6. [9] Mukoyama S, Mukai M, Yasuda S, Oida Y, Himeno S, Nishi T, Nakasaki H, Iwase H, Yasuda M, Sadahiro S, et al. A successfully treated case of severe necrotizing fasciitis
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caused by acute appendicitis: a case report. Tokai J Exp Clin Med 2003 Oct;28(3):139–43. Azab B, Afthinos J, Gibbs KE. Extensive abdominal wall necrotizing fasciitis and fournier's gangrene complicating perforated appendicitis. Gastroenterology 2013;144(5):S1070. Wilharm A, Gras F, Mückley T, Hofmann GO. Necrotizing fasciitis after banal back pain. an unusual course of a retrocoecal appendicitis and its sequellae. Chirurg 2010;81(5):472–6. Ketwong K, Jitpratoom P, Chonlathee P, Anuwong A. Loss of entire abdominal wall in severe malnutrition patient: how i do it? Hernia 2017;21(2):S186. Jacobs P, van der Sluis R, Tack C, Wobbes T. Necrotising fasciitis of the lower limb caused by undiagnosed perforated appendicitis, which necessitated disarticulation of the hip. Eur J Surg 1993 May;159(5):307–8. Ford BM, Tai S, Fantegrossi WE, Prather PL. Synthetic pot: not your grandfather's marijuana. Trends Pharmacol Sci 2017 Mar;38(3):257–76. Tait RJ, Caldicott D, Mountain D, Hill SL, Lenton S. A systematic review of adverse events arising from the use of synthetic cannabinoids and their associated treatment. Clin Toxicol (Phila) 2016;54(1):1–13. Rose DZ, Guerrero WR, Mokin MV, Gooch CL, Bozeman AC, Pearson JM, Burgin WS. Hemorrhagic stroke following use of the synthetic marijuana "spice". Neurology 2015 Sep 29;85(13):1177–9. Aydin S, Yuksel O, Aydin AE, Kizilkilic O, Celik SE. Intracerebral hemorrhage with multiple intracranial arterial stenoses in a synthetic cannabinoid "spice" user. Asian
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Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc
Acute appendicitis complicated by necrotizing fasciitis in a teenager a
a
a
T
b
Jeremy D. Kauffman , Megan O'Brien , Christopher W. Snyder , S. Alex Rottgers , Drew A. Rideouta, Nicole M. Chandlera,∗ a b
Division of Pediatric Surgery, Johns Hopkins All Children's Hospital, 601 5th Street South, Suite 306, St. Petersburg, Florida 33701, USA Division of Plastic and Reconstructive Surgery, Johns Hopkins All Children's Hospital, 601 5th Street South, Suite 306, St. Petersburg, Florida 33701, USA
A R T I C LE I N FO
A B S T R A C T
Keywords: Necrotizing fasciitis Appendicitis Pediatrics Abdominal wall reconstruction Synthetic marijuana
Necrotizing fasciitis is a rare complication of appendicitis in children and is associated with significant morbidity and mortality. We present the case of a 16-year-old male who presented with perforated appendicitis and subsequently developed necrotizing fasciitis of the abdominal wall and perineum. His seven-week hospital course was notable for 16 trips to the operating room, which culminated in staged, complex abdominal wall repair. Ultimately he was discharged home in good condition. The case is remarkable for the extent of soft tissue damage and complexity of repair precipitated by an uncommon complication of a common pediatric disease.
1. Introduction Appendicitis is a common cause of an acute abdomen in children [1]. Delay in treatment increases the likelihood of complications [2]. Necrotizing fasciitis is an uncommon complication of appendicitis, particularly in children, but is associated with significant morbidity and mortality [3]. We present the case of a 16-year-old male who presented with perforated appendicitis, underwent laparoscopic appendectomy, and developed necrotizing fasciitis. The case illustrates the need for timely diagnosis and aggressive surgical management of necrotizing fasciitis, as well as the need for vigilance in working-up even the most routine of pediatric diseases. 2. Case report A 16 year-old, previously healthy male presented to a community hospital with abdominal pain, scrotal swelling, and signs of dehydration and sepsis. The abdominal pain had been present for three days but the patient had delayed seeking medical care because he attributed the pain to withdrawal from synthetic (“spice”) marijuana. He also reported having been struck in the groin during wrestling practice six days prior to presentation. Testicular ultrasound demonstrated a large hydrocele and what appeared to be a bowel-containing right inguinal hernia. Computed tomography (CT) showed evidence of inflammation in the right lower quadrant and extraluminal air extending into the right scrotum (Fig. 1). The differential diagnosis at this time included perforated appendicitis and traumatic perforation of herniated bowel.
Fluid resuscitation and empiric antimicrobial therapy with piperacillintazobactam were initiated. The patient was transferred to a regional hospital where he was taken to the operating room (OR) for diagnostic laparoscopy, which revealed perforated appendicitis and diffuse peritonitis with no evidence of inguinal hernia. Laparoscopic appendectomy with abdominal washout was performed. The scrotum was aspirated percutaneously due to concern for local extension of the infection, but no purulent material was aspirated and no further intervention was performed. The patient's condition initially improved, but he exhibited persistent tachycardia and developed erythema and bullous changes of the right scrotum and inguinal region, for which clindamycin was added to his antibiotic regimen. Multiple sets of blood cultures drawn at this time grew no organisms. He was taken back to the OR on postoperative day five, at which time he was found to have necrotizing fasciitis of the perineum and right groin. Local debridement was performed and a negativepressure wound vacuum applied. Postoperatively, he remained intubated and required ionotropic support. Gram stain of scrotal and abdominal fluid demonstrated gram negative rods and gram positive cocci. Cultures eventually grew Escherichia coli with intermediate resistance to piperacillin-tazobactam, as well as Bacteroides fragilis and Streptococcus constellatus. Meropenem and vancomycin were initiated, and piperacillin-tazobactam was discontinued. Two days later he remained in critical condition and was transferred to a quaternary care pediatric hospital. His APACHE IV score at the time of transfer was 70, with a corresponding mortality risk of 37.3% [4]. He was taken to the OR for debridement of the scrotum, abdominal wall,
∗
Corresponding author. 601 5th St. South, Dept 70-6600, 3rd Floor, St. Petersburg, Florida, 33701 USA. E-mail addresses: jkauff[email protected] (J.D. Kauffman), [email protected] (M. O'Brien), [email protected] (C.W. Snyder), [email protected] (S.A. Rottgers), [email protected] (D.A. Rideout), [email protected] (N.M. Chandler). https://doi.org/10.1016/j.epsc.2018.07.018 Received 17 July 2018; Accepted 21 July 2018 Available online 25 July 2018 2213-5766/ © 2018 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
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Fig. 3. Appearance of wound on hospital day seven following initial four debridements. Surgical concerns at this time included: 1) full thickness abdominal all defect with exposed liver (arrow) and colon (arrowhead) 2) exposed bilateral testicles (circles) and 3) massive skin defect.
complex abdominal wall reconstruction. Control of the infection ultimately required removal of the external oblique, serratus, and inferior latissimus dorsi muscles on the right side, resulting in a right flank wound measuring 68 × 15 cm, right leg wound measuring 30 × 8 cm, and a left flank and perineum wound measuring 30 × 8 cm. The full thickness defect resulted in exposure of the liver, colon, and bilateral testicles (Fig. 3). Reconstruction of the right abdominal wall was achieved in a staged fashion by means of an acellular dermal matrix (Fig. 4), gracilis flap, and vastus lateralis flap (Fig. 5). Repair on the left side was accomplished with primary closure over a drain (Fig. 6). The patient's course was complicated early on by pneumonia, which
Fig. 1. CT at time of initial presentation. Coronal view demonstrating 1) appendicolith (arrow), 2) extraluminal air in the peritoneum and retroperitoneum (arrowheads), and 3) pneumoscrotum (circle).
perineum, right lateral chest wall and flank (Fig. 2). Over the course of the next three weeks he was taken to the OR an additional ten times for wound debridement, negative pressure dressing application, and
Fig. 2. Before (panel A) and after (panels B–D) initial debridement at the children's hospital.
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Fig. 4. Initial closure of right-sided, full-thickness abdominal wall defect with an acellular dermal matrix.
Fig. 6. Partial primary closure of the left perirectus wound over a drain.
Fig. 7. Right flank wound covered with cadaveric skin graft.
Metronidazole was eventually added for better anaerobic coverage, and fluconazole was added when Candida albicans grew from abdominal wound and pleural fluid cultures. Linezolid was added late in his course for growth of Staphylococcus haemolyticus from the left groin wound. Antibiotic and antifungal therapy were ultimately discontinued seven weeks after his initial presentation. Temporary skin closure with cadaver graft was performed (Fig. 7) in order to allow time for nutritional optimization and wound granulation prior to definitive closure with split-thickness skin grafting. Nine weeks and five days after his initial presentation, he was discharged home. When he returned to clinic for follow-up four weeks later, his wounds were healing well with less than one percent total surface area of breakdown throughout the skin graft (Fig. 8). With the exception of a brief admission for drainage of an abscess of the medial right thigh, the patient has continued to progress well and has returned to his normal activities of daily living. Fig. 5. Reconstruction of the (A) right abdominal wall with acellular dermal matrix and gracilis flap and (B) vastus lateralis muscle flap mobilized and reflected proximally to cover right flank mesh repair.
3. Discussion The case described above is notable on several accounts. First, necrotizing fasciitis is an uncommon complication of appendicitis, particularly in the pediatric population [5,6]. In our review of the literature, which was not restricted by language and included searches in PubMed, Ovid MEDLINE, and Embase, we identified a total of 42 cases (Appendix 1). Median age of the cohort was 65 years (IQR = 44–77).
improved following treatment with parenteral antibiotics and antifungals. Later in his course he developed an enterocutaneous fistula, which resolved with conservative therapy. Meropenem was continued throughout his course, while vancomycin was discontinued shortly after his third operation with concern for worsening acute renal failure.
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Fig. 8. Wound appearance at one month follow-up.
and even a diagnosis as routine as appendicitis must be considered judiciously.
Only two cases occurred in children. One of these occurred in a 19-dayold male who developed necrotizing fasciitis following appendectomy and subsequently died of multiple organ dysfunction [7]. The second occurred in a ten-year-old male who required repair with a skin graft and survived [8]. When considering patients of all ages identified in our review, in-hospital mortality was 35.1%, and median hospital length of stay was six weeks (IQR = 2–11) among those who survived. The case is also notable for the extent of tissue loss and associated complexity of reconstructive repair. While several reports of necrotizing fasciitis following appendicitis describe repair of the abdominal wall with skin grafts, one abstract mentions the use of regional advancement flaps, and one article describes disarticulation of the hip, we encountered no report of reconstruction with gracilis and vastus lateralis muscle flaps [9–13]. The case demonstrates that excellent functional outcomes can be achieved in the setting of full-thickness loss of portions of the abdominal and perineal wall using a combination of local wound care and staged reconstruction with biologic and autologous grafts. Perhaps of only incidental consequence is the temporal association of synthetic (“spice”) marijuana use and the onset of the patient's initial symptoms. It may be worth noting that synthetic marijuana has been associated with a plethora of untoward side effects, ranging from mild to potentially lethal in severity [14,15]. These include such “bad actors” as seizure, stroke, myocardial infarction, and cardiogenic shock, in addition to the “usual suspects” of vomiting, confusion, and psychosis [16–19]. To our knowledge there have been no reports of appendicitis or necrotizing fasciitis in association with synthetic marijuana. While there is insufficient evidence to support a causal relationship between spice use and our patient's pathology, the patient's perception that his initial symptoms were due to spice withdrawal contributed to a delay in treatment, which in turn resulted in greater morbidity.
Author contribution
• Jeremy Kauffman: Study design, drafting and critical revision of the manuscript, statistical analysis, literature review. • Megan O'Brien: Study design, drafting and critical revision of the manuscript, literature review. • Christopher W. Snyder: Study design, photography, critical revision of the manuscript • S. Alex Rottgers: Study design, photography, critical revision of the manuscript • Drew A. Rideout: Data interpretation, critical revision of the manuscript • Nicole Chandler: Study design, critical revision of the manuscript. Institutional review board The Johns Hopkins Medicine IRB does not require IRB approval for case reports. Therefore IRB approval to publish this care report was not obtained. Patient consent Witten consent of the patient's mother and written assent of the patient to publish this case report were obtained. Funding No funding or grant support.
4. Conclusion
Authorship
In conclusion, this case highlights the fact that necrotizing infection can lead to rapid and extensive soft tissue loss with potentially lifethreatening consequences in children with appendicitis. Timely detection and expeditious triage to an appropriate level of care are critical to survival in these circumstances. Early and aggressive debridement of necrotic tissue, rapid initiation of broad-spectrum antibiotics, and assiduous supportive care with appropriate respiratory and hemodynamic support are the mainstay of treatment [20]. This case demonstrates that children are not immune to the lethal risks posed by this complication,
All authors attest that they meet the current ICMJE criteria for Authorship. Conflict of interest The following authors have no financial disclosures: JDK, MO, CWS, SAR, DAR, NMC.
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Appendix 1. Cases of Appendicitis complicated by necrotizing fasciitis reported in the literature.
No.
Author
Year
Sex
Age∗
In-Hospital Mortality
Length of Stay∗∗
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31–33 34 35 36 37 38 39 40 41 42
Awe[1] Azab[2] Bobrow[3] Cano[4] Chen[5] Coulier[6] Fernandes[7] Fujiwara[8] Garcia-Santos[9] Georgiou[10] Gerber[11] Groth[12] Guirguis[13] Hartley[14] Harwant[15] Hensley[16] Hua[17] Huang[18] Hung[19] Hung[19] Jacobs[20] Ketwong[21] Kjossev[22] Lodha[23] Mai[24] Marinis[25] Marron[26] Mazza[27] Mukoyama[28] Nazir[29] Oztas[30] Penninga[31] Raghavendra[32] Rajaguru[33] Romanoff[34] Şenkul[35] Taif[36] Takeda[35] Tsimogianni[37] Wilharm[38]
2003 2013 1996 2016 2010 2012 2011 1999 2012 2013 1994 1999 1989 1994 2001 2011 2015 2018 2015 2015 1993 2017 2003 2003 2010 2015 2005 1987 2003 2012 2013 2004 2010 2016 2016 2003 2014 2012 2012 2010
Male Male Male Female Female Male Male Female Male Male Male Female Female Female Female Female Male Male Male Female Female Female Female Male Male Female Female Female Male Female NR Female Male Male Male NR Female Male Male Male
28 23 63 62 76 41 10 98 80 84 85 49 80 79 66 69 50 65 73 91 44 63 83 0.05 65 83 67 59 77 72 NR 33 19 47 71 NR 26 76 52 21
No No Yes No No No No No NR No No No NR Yes Yes No Yes Yes No Yes No No Yes Yes No Yes No Yes No No NR No No No No Yes Yes Yes No No
33 60 NA 45 73 NR NR 16 NR 22 NR NR NR NA NA 28 NA NA 60 NA 40 120 NA NA 14 NA NR NA 80 NR NR NR NR NR NR NA NA NA NR 91
NR - Not reported; NA - Not applicable. * Age in years. ** Length of stay in days.
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