- Email: [email protected]
Schistosoma ova found in gastrostomy site granulation tissue
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports
Schistosoma ova found in gastrostomy site granulation tissue Muhammad Elsayed Mahmoud a, c, *, Khaled Zamel Aldaraan a, Mohamed Hany Hassab a, Sana Faraj Aljabr b Pediatric Surgery Department, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia Colorectal Service, Stoma Nursing Care, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia c Pediatric Surgery Department, Faculty of Medicine, Al-Azhar University, Cairo, Egypt a
b
ARTICLE INFO
ABSTRACT
Keywords: Severe traumatic brain injury Gastrostomy Laparoscopic gastrostomy Schistosoma infestation Silver nitrate application
Background: Severe traumatic brain injury (TBI) occurs rarely in children. Gastrostomy tube placement is a frequently performed procedure in children and has various indications including neurologic impairment. One of the common complications of gastrostomy is granulation tissue formation resulting from chronic irritation. Case presentation: We present the case of 12-years-old Asian male patient that was referred to our hospital as a victim of road traffic accident (RTA) with polytrauma (severe traumatic brain injury, fracture left tibia & fibula). He underwent laparoscopic gastrostomy for long-term feeding. Post tube placement, granulation tissue formed and persisted in spite of multiple silver nitrate cauterization trials necessitating surgical removal. On histopathologic examination of the excised specimen, it was found to harbour schistosoma ova. Conclusion: Gastrostomy tube placement is commonly performed in pediatric cases with serious neurologic insult. Many cases of Schistosoma infestation are asymptomatic. Some cases have atypical presentation including histopathologic diagnosis.
1. Introduction Head injury is an increasingly common cause of emergency department (ED) visits for pediatric patients and is considered a leading cause of mortality, morbidity and residual neurologic deficits. In the United States, pediatric traumatic brain injury (TBI) led to more than 2.5 million visits and 50,000 hospitalizations per year. Deaths from pediatric TBI are most common in the adolescent from road traffic accidents (RTA) & motor vehicle collisions (MVC) and in children younger than 4 years from falls [1,2]. Severe or clinically important traumatic brain injury is mainly caused by blunt trauma and its indicators include lethal head injury, need for neurosurgical intervention, prolonged intubation, long hospital stay, intensive care unit (ICU) admission, and the likely to develop significant long term neurologic impairment [1]. Gastrostomy is a commonly performed procedure in children. It has several indications involving failure to thrive, swallowing disorders, neurological impairment, severely traumatized children and malnutrition for purpose of long-term enteral feeding. About 11–26%
*
of children with gastrostomy tubes will experience complications during the early course of their care [3–5]. Schistosomaiasis is a common parasitic disease and is also a major public health problem in many parts of the developing world, especially in sub-Saharan Africa, Western Asia, and Southeast Asia. The disease is caused by blood flukes (trematode worms) of the genus Schistosoma, with Schistosoma haematobium, Schistosoma mansoni, and Schistosoma japonicum triggering most infections [6,7]. Although schistosomiasis is one of the most common helminth infections worldwide, it is poorly documented in Asia. Schistosomiasis and other soil-transmitted helminths are chronic diseases but case detection is difficult because most cases are asymptomatic. However, schistosomiasis is endemic in Asia especially poor countries and its burden is significant [8]. To our knowledge, no previous pediatric cases of schistosoma infested stomach or gastrostomy site have been similarly reported. Herein, we present a case of critically traumatized patient who underwent laparoscopic gastrostomy complicated by granulation tissue formation which was surgically excised and had been found to harbour parasitic (schistosoma) ova.
Corresponding author. Pediatric Surgery Department, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia.; E-mail address: [email protected], [email protected] (M.E. Mahmoud).
https://doi.org/10.1016/j.epsc.2019.101377 Received 8 December 2019; Received in revised form 16 December 2019; Accepted 20 December 2019 Available online 26 December 2019 2213-5766/© 2019 The Authors. Published by Elsevier Inc. This is an open (http://creativecommons.org/licenses/by-nc-nd/4.0/).
access
article
under
the
CC
BY-NC-ND
license
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
2. Case
longed intubation to facilitate long term respiratory care. He was fed through nasogastric tube (NGT). Tracheostomy was closed 3.5 months later after confirmation of safe decannulation by normoxemia on room air, no desaturation, & no respiratory distress. Closure of the tracheostomy resulted in improvement of subglottic closing pressure necessary for effective swallowing. He underwent laparoscopic gastrostomy placement for long term enteral feeding 3 months after trauma. Intra-operatively, the transverse colon was found intervening between the stomach and anterior abdominal wall, so it was wise & fortunately to be done by the laparoscopic technique otherwise it could result in gastrocolocutaneous fistula. Due to improper nursing care, the gastrostomy tube was dislodged twice post-operatively and successfully re-inserted with no complications of tube catheter change such as tract disruption, misplacement, or trauma. The tube also migrated internally several times with impaction of the tube bifurcation in the wound for long time resulting in irritation and granulation tissue formation (Fig. 2). Trials to ablate the granulation tissue by topical application of silver nitrate sticks were done over 6 weeks but unfortunately failed and also discolored the skin (Fig. 3) thus mandating surgical excision and closure of the gastrostomy as the patient no longer required it. Oral feeding markedly improved 3 months post placement (after documentation of safe effective oral intake by improved modified Barium swallowing study
A 12-years-old male patient was brought by the Red Crescent to the ED of our hospital as a victim of RTA. Primary survey revealed that he was comatosed with Glasgow coma scale (GCS) 5/15, intubated by the emergency medical service (EMS) personnel, ventilated, intact patent airway, equal bilateral air entry, tachycardic (122 beat per minute bpm), normotensive 140/72 mmHg, left flank abrasion, and negative extended focussed sonography for trauma (E-FAST). Secondary survey showed left lower chest abrasion & emphysema, left leg deformed and splinted due to left tibia & fibula closed fracture with intact peripheral pulse and no signs of compartment syndrome and digital rectal examination revealed lost tone with loaded rectum. Foley's catheter was inserted and revealed clear urine. Laboratory studies revealed anemia, eosinophilia, monocytosis, & basophilia. Hypoalbuminemia also was found and eventually improved. Non-enhanced computerized tomography CT trauma scan was nearly normal at first but on repetition, it revealed right frontoparietal subgaleal hematoma, intraventricular hemorrhage seen within the right lateral ventricle, third ventricle, and interpeduncular cistern. Also, CT scan revealed focal hyperdensity within the left hypothalamus/cerebral peduncle likely blood, innumerable hemorrhagic foci in grey-white matter interface of bilateral cerebral hemispheres, left cerebral peduncle, bilateral thalami more on right side, left basal ganglia, pons, & right cerebellar hemisphere with diffuse axonal injury mainly affecting the body and splenium of the corpus callosum all giving a picture suggestive of severe traumatic brain injury (TBI). CT chest revealed right upper lobe subcutaneous emphysema, small left lower lung contusion & bilateral basal lung atelectasis. CT abdomen revealed intact viscera with mild periportal edema. Plain x-ray skeleton revealed left tibia and fibula closed fracture (Fig. 1). The patient was resuscitated and prepared for urgent surgical intervention. The patient underwent external ventricular drainage and evacuation of the intracranial hemorrhage by neurosurgeons team. Postoperatively, he was admitted at pediatric intensive care unit (PICU), intubated, sedated, and mechanically ventilated. The EVD and intracranial pressure (ICP) monitoring device were removed 10 days post-operatively. One week later after stabilization of the patient's general condition, he underwent open reduction & internal fixation by flexible nails for left tibia fracture by orthopedic team and tracheostomy by ENT team for anticipated long PICU stay with pro-
Fig. 2. Granulation tissue at the gastrostomy edge.
Fig. 1. X-ray left lower limb showed fracture leg both bones (blue lines). (For interpretation of the references to color in this figure legend, the reader is referred to the Web version of this article).
Fig. 3. Skin discoloration caused by silver nitrate. 2
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
(MBSS). Two tiny mucosal pits remained and successfully cauterized by silver nitrate as shown in Fig. 4. On histopathologic examination of the excised granulation tissue, schistosoma ova were found although no history of schistosomiasis was obtained indicating subclinical infestation (Figs. 5 and 6). The pathology report stated that sections represent inflamed granulation tissue composed of organized blood vessels and collagen fibers cov-
ered partially by benign squamous epithelium with acute and chronic inflammatory infiltrates with few scattered parasitic eggs favoring schistosoma ova (elliptical shape with terminal spike consistent with Schistosoma hematobium species). Upon this discovery, urine and stool analysis were done and revealed negative and serology for Schistosoma was done and revealed positive and the patient had been given anthelminthic medication (praziquantel). The primary trauma resulted in right spastic hemiparesis, speech dysarthria, and mild-to-moderate receptive and expressive aphasia and cognitive impairment. The patient underwent bilateral percutaneous tendon Achilles lengthening tenotomy (TAL) 7.5 months post trauma to treat spastic equinovarus deformity and Botox injection in right arm muscles 1 month later to manage the tight muscles with marked improvement of mobility. The patient's mobility, speech status, and neurocognitive ability were markedly improved after long-term follow up with physiotherapy, occupational therapy, rehabilitation, speech therapy & neuropsychiatry. 3. Discussion The availability of computed tomography (CT) scan as essential setup in ED enables physicians to readily diagnose brain injuries with high diagnostic accuracy. Once diagnosed, the site, size, course of injury, management plan are determined and also prognosis can be expected. Proper management of traumatic brain injury mainly depends on identification of primary injury and prevention of the secondary injury that essentially caused by hypoxia and hypotension [9–12]. Also, hypothermia, hypocapnia, hypoglycaemia and increased intracranial pressure are independent predictors of mortality [1,13]. Disability assessment generally uses the Glasgow Coma Scale (GCS) to stratify the risk into mild [13–15], moderate [9–12], and severe [3–8] head injury. Early initiation of therapy and the use of standardized guidelines in the management of moderate and severe TBI are essential to improve outcomes in pediatric patients [1,14]. Enteral access via gastrostomy tube is a common procedure in children. The laparoscopic approach to gastrostomy tube placement has been advocated by numerous centres. Several studies have compared percutaneous endoscopic gastrostomy (PEG) to laparoscopic gastrostomy (LG) placement in children finding the latter to have longer operative times but less serious intra- & post-operative complications [15,16]. With demonstration of safe early feeding after PEG, it seems to be a safe reasonable practice to initiate post-operative feeding as early as 4 h in patient undergoing laparoscopic gastrostomy with most patients tolerated with low risk for adverse events [16]. Laparoscopic gastrostomy is flourishing minimally invasive technique for gastrostomy tube placement in children. Based on advantages of the laparoscopic approach as direct visualization of the abdominal cavity, minimize unintentional hollow viscera injury as colon perforation, transfascial tacking suture to fix the tube, reduce risk of early tube dislodgement, & obviate the need to exchange tube for skin-level, balloon-based tube type, multiple studies have adopted for LG as the procedure of choice for children [16–21] as in our case. The gastrostomy was fixed to posterior rectus sheath and fascia by nonabsorbable sutures under vision. This method of tube fixation offers the least risk of tube dislodgement but increased surgical site infection rate [5,22]. Our patient was cared for by many other specialities as neurosurgery, rehabilitation, physiotheraspy, occupational therapy, speech, and pediatric neuropsychiatry as in Riachard et al., 2018 study [16]. The common complications of gastrostomy tube placement include surgical site infection, tube dislodgement, granulation tissue formation, peristomal leakage, stenosis, retraction, & proximal or distal tube migration [5]. Investigators have proposed different definitions and
Fig. 4. Two tiny mucosal pits remained and successfully cauterized by silver nitrate (green lines). (For interpretation of the references to color in this figure legend, the reader is referred to the Web version of this article).
Fig. 5. Schistosoma hematobium ova found in the excised granulation tissue.
Fig. 6. Schistosoma hematobium ova found in the excised granulation tissue. 3
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
categories for gastrostomy complications into either pre-procedural, intra-procedural or post-procedural, either early, or late, minor (approximately 23.6%) or major (about 8.8%), procedure-related, tube related, stoma-related, or related to patient feeding intolerance or change in clinical status as worsened gastroesophageal reflux disease (GERD) or aspiration pneumonia [5,21]. Granulation tissue has many nomenclatures as proposed by several authors. Some call it granulation tissue; others call it overgrowth of gastric mucosa or pseudotumour gastric mucosa as mentioned in the histopathology report [23]. The prevention and management of overgranulation around gastrostomy sites is a challenge for nurses to manage. Harris et al. [24] defines 3 types of overgranulation, Type one considers that overgranulation is caused by inflammatory factors that may be caused by an irritant at the wound edge. Type two is caused by moist environment. Type three is caused by cellular imbalance and has no specific treatment options other than to eradicate type 1 and 2 causation factors. Tenaka et al. [25] found that sprinkling salt reduced or removed gastrostomy overgranulation because salt draws water out of the edematous tissue, across the cell membrane, removing fluid and possible contaminants into the salt crystals [23]. Schiostsoma ova may be the causative factor (by chronic irritation) of granulation tissue formation. It reached the stomach via submucosal blood vessels from porto-mesenetric circulation or the mesentery if it is of Schistosoma mansoni type but in our case, no peristomal mesenteric hernia occurred and we hypothesize that the Schistosoma haematobium ova reached the stomach via retrograde manner from the urinary bladder retroperitoneal venous system or may be via still patent porto-systemic anastomosis. The disease control strategy for schistosomiais in Asia comprised three main approaches: (i) control of the snail intermediate host; (ii) treatment of all infected people; and (iii) concreting over the wetland habitat of the snail host. Existing control programs in endemic areas aimed at improving water supply, sanitation, toilet facilities, and hygiene such as healthy lifestyle & cultivation techniques have led to decreased infection rates [8]. Deworming is a preventive control measure but, to keep reinfection rates as low as possible, preventive education must be implemented widely and continuously throughout the region particularly in rural and remote communities in developing countries [8,26,27]. Schistosomiasis is transmitted by direct percutaneous penetration by cercariae, the free-swimming larval form of the worm, on exposure to infected fresh water snails. More than 200–240 million people world-wide are affected especially children in pre-school and school age groups, fishermen, and farmers. Commonly, these infections have subclinical symptoms in children and young adults [27]. Symptomatic infected cases presented firstly by maculpapular rash then after several weeks when the adult worms develop and start to lay eggs, the host defence reacts to the deposited ova leading to fever, headache, myalgia, hematuria, tender hepatosplenomegally, genitourinary tract disease, and bowel & hepatic fibrosis [28]. Anemia, nutritional deficiencies and stunted growth & cognitive delays are observed in chronic disease [29]. Diagnosis can be achieved by finding parasite ova in urine or stool and by serologic testing. On microscopy, the spike orientation helps in determination of species. Other supportive laboratory findings include eosinophilia, anemia, hypoalbuminemia [30]. The world health organization (WHO) aims at elimination of schistosomiasis globally by year 2025 [31]. The disease control program includes mass drug administration (MDA) or what is called preventive chemotherapy of praziquantel 40 mg/kg one dose in endemic areas given to high risk populations mainly preschool and school age children, adolescent girls, fishermen, farmers, and house-wife women. Also, improving access to drinking water, sanitation, hygiene, bovine treatment, snail control, modern irrigation & cultivation techniques, deworming strategies, and imple-
menting health education programmes with the community targeting the aforementioned high risk groups associated with annual MDA should be utilized in order to interrupt the worm life cycle and to achieve sustainable disease control [26,27]. 4. Conclusion Gastrostomy is one of the commonly performed procedures in children. It has several indications as swallowing disorders, failure to thrive, malnutrition, neurologic insult/impairment and various techniques including open, laparoscopic, endoscopic, or radiographic assisted. Schistosoma infestation can manifest in several ways but strangely the stomach. This mode of presentation should be considered to help diagnosis and to shed light spot on proper hygiene, infection prevention & control. Patient consent This report does not contain any personal information that could lead to the identification of the patient. Funding No funding or grant resources to disclose. Authorship All authors attest that they meet the current ICMJE criteria for Authorship. Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. List of abbreviations bpm CT ED E-FAST EMS EVD GCS GERD ICP ICU LG MBBS MDA MVC NGT PEG PICU RTA TAL TBI WHO
beat per minute Computerized tomography Emergency department Extended focussed assessment with sonography for trauma Emergency medical service External ventricular drainage Glasgow coma scale Gastroesophageal reflux disease Intracranial pressure Intensive care unit Laparoscopic gastrostomy Modified barium swallowing study Mass drug administration Motor vehicle collision Nasogastric tube Percutaneous endoscopic gastrostomy Pediatric intensive care unit Road traffic accident Tendon Achilles lengthening Traumatic brain injury World health organization
Appendix A. Supplementary data Supplementary data to this article can be found online at https:// doi.org/10.1016/j.epsc.2019.101377.
4
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
References
[16] Hendrickson RJ, Poola AS, Sujka JA, Weaver KL, Rentea RM, Peter SDS, et al. Feeding advancement and simultaneous transition to discharge (FASTDischarge) after laparoscopic gastrostomy. J Pediatr Surg 2018;53:2326–30. [17] Zamakhshary M, Jamal M, Blair GK, Murphy JJ, Webber EM, Skarsgard ED. Laparoscopic vs percutaneous endoscopic gastrostomy tube insertion: a new pediatric gold standard?. J Pediatr Surg 2005;40(5):859–62. [18] Akay B, Capizzani TR, Lee AM, Drongowski RA, Geiger JD, Hirschl RB, et al. Gastrostomy tube placement in infants and children: is there a preferred technique?. J Pediatr Surg 2010;45:1147–52. [19] Richards MK, Li CI, Foti JL, Leu MG, Wahbeh GT, Shaw D, et al. Resource utilization after implementing a hospital-wide standardized feeding tube placement pathway. J Pediatr Surg 2016;51:1674–9. [20] Sunstrom R, Hamilton N, Fialkowski E, Lofberg K, McKee J, Sims T, et al. Minimizing variance in pediatric gastrostomy: does standardized perioperative feeding plan decrease cost and improve outcomes?. Am J Surg 2016;211:948–53. [21] McSweeney ME, Smithers CJ. Advances in pediatric gastrostomy placement. Gastrointest Endosc Clin N Am 2016;26(1):169–85. [22] Aprahamian CJ, Morgan TL, Harmon CM, Georgeson KE, Barnhart DC. U-stitch laparoscopic gastrostomy technique has a low rate of complications and allows primary button placement: experience with 461 pediatric procedures. Laparoendosc Adv Surg Tech A 2006;16:643–9. [23] Trautner E, Booth L. An audit around the use of salt in the treatment of overgranulation around PEG sites. Clin Nutr ESPEN 2018;28:273–4. [24] Harris A, Rolstad B. Hypergranulation tissue: a non-traumatic method of management. Ostomy Wound Manage 1994;40(5):20–30. [25] Tenaka H, Arai K, Fujino A. Treatment for hypergranulation at gastrostomy sites with sprinkling salt in pediatric patients. J Wound Care 2013;22(1):17–8. 20. [26] Yu W, Ross AG, Olveda RM, Harn DA, Li Y, Delia C, et al. Risk of human helminthiases: geospatial distribution and targeted Control. Int J Infect Dis 2017; 55:131–8. [27] Weatherhead JE, Hotez PJ, Mejia R. The global state of helminth control and elimination in children. Pediatr Clin N Am 2017;64:867–77. [28] Ross AG, Bartley PB, Sleigh AC, Olds GR, Li Y, Williams GM, et al. Schistosomiasis. N Engl J Med 2002;346(16):1212–20. [29] Hotez PJ, Fenwick A. Schistosomiasis in Africa: an emerging tragedy in our new global health decade. PLoS Neglected Trop Dis 2009;3(9):e485. [30] Gray DJ, Ross AG, Li YS, McManus DP. Diagnosis and management of schistosomiasis. BMJ 2011;342:d2651. [31] Hansen C, Paintsil E. Infectious diseases of poverty in children: a tale of two worlds. Pediatr Clin N Am 2016;63:37–66.
[1] Leetch AN, Wilson B. Pediatric major head injury not a minor problem. Emerg Med Clin N Am 2018;36:459–72. [2] Taylor CA. Traumatic brain injury–related emergency department visits, hospitalizations, and deaths—United States, 2007 and 2013. MMWR Surveill Summ 2017;66:1–16. [3] Pearce CB, Duncan HD. Enteral feeding. Nasogastric, nasojejunal, percutaneous endoscopic gastrostomy, or jejunostomy: its indications and limitations. Postgrad Med J 2002;78:198–204. [4] Pemberton J, Frankfurter C, Bailey K, Jones L, Walton JM. Gastrostomy matters−the impact of pediatric surgery on caregiver quality of life. J Pediatr Surg 2013;48:963–70. [5] Mason CA, Skarda DE, Bucher BT. Outcomes after laparoscopic gastrostomy suture techniques in children. J Surg Res 2018;232:26–32. [6] Utzinger J, Raso G, Brooker S, De Savigny D, Tanner M, Ornbjerg N, et al. Schistosomiasis and neglected tropical diseases: towards integrated and sustainable control and a word of caution. Parasitology 2009;136:1859–74. [7] Coulibaly JT, Panic G, Silué KD, Jana K, Jan H, Keiser J. Efficacy and safety of praziquantel in preschool-aged and school-aged children infected with Schistosoma mansoni: a randomised controlled, parallel-group, dose-ranging, phase 2 trial. Lancet Glob Health 2017;5:e688–98. [8] Ohta N, Waikagul J. Disease burden and epidemiology of soil-transmitted helminthiases and schistosomiasis in Asia: the Japanese perspective. J Parasit Trends 2006;23(1):30–5. [9] Adelson P. Guidelines for the acute medical management of severe traumatic brain injury in infants, children, and adolescents. Pediatr Crit Care Med 2003;4(3): 1–75. [10] McHugh GS, Engel DC, Butcher I, Steyerberg EW, Lu J, Mushkudiani N, et al. Prognostic value of secondary insults in traumatic brain injury: results from the IMPACT study. J Neurotrauma 2007;24(2):287–93. [11] Chang W-TW, Badjatia N. Neurotrauma. Emerg Med Clin N Am 2014;32(4):889– 905. [12] Spaite DW, Hu C, Bobrow BJ, Chikani V, Barnhart B, Gaither JB, et al. The effect of combined out-of-hospital hypotension and hypoxia on mortality in major traumatic brain injury. Ann Emerg Med 2017;69(1):62–72. [13] Wing R, James C. Pediatric head injury and concussion. Emerg Med Clin N Am 2013;31(3):653–75. [14] Blyth BJ, Bazarian JJ. Traumatic alterations in consciousness: traumatic brain injury. Emerg Med Clin N Am 2010;28(3):571–94. [15] Lantz M, Hultin Larsson H, Arnbjörnsson E. Literature review comparing laparoscopic and percutaneous endoscopic gastrostomies in a pediatric population. Int J Pediatr 2010;e507616.
5
Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports
Schistosoma ova found in gastrostomy site granulation tissue Muhammad Elsayed Mahmoud a, c, *, Khaled Zamel Aldaraan a, Mohamed Hany Hassab a, Sana Faraj Aljabr b Pediatric Surgery Department, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia Colorectal Service, Stoma Nursing Care, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia c Pediatric Surgery Department, Faculty of Medicine, Al-Azhar University, Cairo, Egypt a
b
ARTICLE INFO
ABSTRACT
Keywords: Severe traumatic brain injury Gastrostomy Laparoscopic gastrostomy Schistosoma infestation Silver nitrate application
Background: Severe traumatic brain injury (TBI) occurs rarely in children. Gastrostomy tube placement is a frequently performed procedure in children and has various indications including neurologic impairment. One of the common complications of gastrostomy is granulation tissue formation resulting from chronic irritation. Case presentation: We present the case of 12-years-old Asian male patient that was referred to our hospital as a victim of road traffic accident (RTA) with polytrauma (severe traumatic brain injury, fracture left tibia & fibula). He underwent laparoscopic gastrostomy for long-term feeding. Post tube placement, granulation tissue formed and persisted in spite of multiple silver nitrate cauterization trials necessitating surgical removal. On histopathologic examination of the excised specimen, it was found to harbour schistosoma ova. Conclusion: Gastrostomy tube placement is commonly performed in pediatric cases with serious neurologic insult. Many cases of Schistosoma infestation are asymptomatic. Some cases have atypical presentation including histopathologic diagnosis.
1. Introduction Head injury is an increasingly common cause of emergency department (ED) visits for pediatric patients and is considered a leading cause of mortality, morbidity and residual neurologic deficits. In the United States, pediatric traumatic brain injury (TBI) led to more than 2.5 million visits and 50,000 hospitalizations per year. Deaths from pediatric TBI are most common in the adolescent from road traffic accidents (RTA) & motor vehicle collisions (MVC) and in children younger than 4 years from falls [1,2]. Severe or clinically important traumatic brain injury is mainly caused by blunt trauma and its indicators include lethal head injury, need for neurosurgical intervention, prolonged intubation, long hospital stay, intensive care unit (ICU) admission, and the likely to develop significant long term neurologic impairment [1]. Gastrostomy is a commonly performed procedure in children. It has several indications involving failure to thrive, swallowing disorders, neurological impairment, severely traumatized children and malnutrition for purpose of long-term enteral feeding. About 11–26%
*
of children with gastrostomy tubes will experience complications during the early course of their care [3–5]. Schistosomaiasis is a common parasitic disease and is also a major public health problem in many parts of the developing world, especially in sub-Saharan Africa, Western Asia, and Southeast Asia. The disease is caused by blood flukes (trematode worms) of the genus Schistosoma, with Schistosoma haematobium, Schistosoma mansoni, and Schistosoma japonicum triggering most infections [6,7]. Although schistosomiasis is one of the most common helminth infections worldwide, it is poorly documented in Asia. Schistosomiasis and other soil-transmitted helminths are chronic diseases but case detection is difficult because most cases are asymptomatic. However, schistosomiasis is endemic in Asia especially poor countries and its burden is significant [8]. To our knowledge, no previous pediatric cases of schistosoma infested stomach or gastrostomy site have been similarly reported. Herein, we present a case of critically traumatized patient who underwent laparoscopic gastrostomy complicated by granulation tissue formation which was surgically excised and had been found to harbour parasitic (schistosoma) ova.
Corresponding author. Pediatric Surgery Department, Prince Mohammed bin Abdulaziz Hospital (PMAH), Riyadh, Saudi Arabia.; E-mail address: [email protected], [email protected] (M.E. Mahmoud).
https://doi.org/10.1016/j.epsc.2019.101377 Received 8 December 2019; Received in revised form 16 December 2019; Accepted 20 December 2019 Available online 26 December 2019 2213-5766/© 2019 The Authors. Published by Elsevier Inc. This is an open (http://creativecommons.org/licenses/by-nc-nd/4.0/).
access
article
under
the
CC
BY-NC-ND
license
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
2. Case
longed intubation to facilitate long term respiratory care. He was fed through nasogastric tube (NGT). Tracheostomy was closed 3.5 months later after confirmation of safe decannulation by normoxemia on room air, no desaturation, & no respiratory distress. Closure of the tracheostomy resulted in improvement of subglottic closing pressure necessary for effective swallowing. He underwent laparoscopic gastrostomy placement for long term enteral feeding 3 months after trauma. Intra-operatively, the transverse colon was found intervening between the stomach and anterior abdominal wall, so it was wise & fortunately to be done by the laparoscopic technique otherwise it could result in gastrocolocutaneous fistula. Due to improper nursing care, the gastrostomy tube was dislodged twice post-operatively and successfully re-inserted with no complications of tube catheter change such as tract disruption, misplacement, or trauma. The tube also migrated internally several times with impaction of the tube bifurcation in the wound for long time resulting in irritation and granulation tissue formation (Fig. 2). Trials to ablate the granulation tissue by topical application of silver nitrate sticks were done over 6 weeks but unfortunately failed and also discolored the skin (Fig. 3) thus mandating surgical excision and closure of the gastrostomy as the patient no longer required it. Oral feeding markedly improved 3 months post placement (after documentation of safe effective oral intake by improved modified Barium swallowing study
A 12-years-old male patient was brought by the Red Crescent to the ED of our hospital as a victim of RTA. Primary survey revealed that he was comatosed with Glasgow coma scale (GCS) 5/15, intubated by the emergency medical service (EMS) personnel, ventilated, intact patent airway, equal bilateral air entry, tachycardic (122 beat per minute bpm), normotensive 140/72 mmHg, left flank abrasion, and negative extended focussed sonography for trauma (E-FAST). Secondary survey showed left lower chest abrasion & emphysema, left leg deformed and splinted due to left tibia & fibula closed fracture with intact peripheral pulse and no signs of compartment syndrome and digital rectal examination revealed lost tone with loaded rectum. Foley's catheter was inserted and revealed clear urine. Laboratory studies revealed anemia, eosinophilia, monocytosis, & basophilia. Hypoalbuminemia also was found and eventually improved. Non-enhanced computerized tomography CT trauma scan was nearly normal at first but on repetition, it revealed right frontoparietal subgaleal hematoma, intraventricular hemorrhage seen within the right lateral ventricle, third ventricle, and interpeduncular cistern. Also, CT scan revealed focal hyperdensity within the left hypothalamus/cerebral peduncle likely blood, innumerable hemorrhagic foci in grey-white matter interface of bilateral cerebral hemispheres, left cerebral peduncle, bilateral thalami more on right side, left basal ganglia, pons, & right cerebellar hemisphere with diffuse axonal injury mainly affecting the body and splenium of the corpus callosum all giving a picture suggestive of severe traumatic brain injury (TBI). CT chest revealed right upper lobe subcutaneous emphysema, small left lower lung contusion & bilateral basal lung atelectasis. CT abdomen revealed intact viscera with mild periportal edema. Plain x-ray skeleton revealed left tibia and fibula closed fracture (Fig. 1). The patient was resuscitated and prepared for urgent surgical intervention. The patient underwent external ventricular drainage and evacuation of the intracranial hemorrhage by neurosurgeons team. Postoperatively, he was admitted at pediatric intensive care unit (PICU), intubated, sedated, and mechanically ventilated. The EVD and intracranial pressure (ICP) monitoring device were removed 10 days post-operatively. One week later after stabilization of the patient's general condition, he underwent open reduction & internal fixation by flexible nails for left tibia fracture by orthopedic team and tracheostomy by ENT team for anticipated long PICU stay with pro-
Fig. 2. Granulation tissue at the gastrostomy edge.
Fig. 1. X-ray left lower limb showed fracture leg both bones (blue lines). (For interpretation of the references to color in this figure legend, the reader is referred to the Web version of this article).
Fig. 3. Skin discoloration caused by silver nitrate. 2
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
(MBSS). Two tiny mucosal pits remained and successfully cauterized by silver nitrate as shown in Fig. 4. On histopathologic examination of the excised granulation tissue, schistosoma ova were found although no history of schistosomiasis was obtained indicating subclinical infestation (Figs. 5 and 6). The pathology report stated that sections represent inflamed granulation tissue composed of organized blood vessels and collagen fibers cov-
ered partially by benign squamous epithelium with acute and chronic inflammatory infiltrates with few scattered parasitic eggs favoring schistosoma ova (elliptical shape with terminal spike consistent with Schistosoma hematobium species). Upon this discovery, urine and stool analysis were done and revealed negative and serology for Schistosoma was done and revealed positive and the patient had been given anthelminthic medication (praziquantel). The primary trauma resulted in right spastic hemiparesis, speech dysarthria, and mild-to-moderate receptive and expressive aphasia and cognitive impairment. The patient underwent bilateral percutaneous tendon Achilles lengthening tenotomy (TAL) 7.5 months post trauma to treat spastic equinovarus deformity and Botox injection in right arm muscles 1 month later to manage the tight muscles with marked improvement of mobility. The patient's mobility, speech status, and neurocognitive ability were markedly improved after long-term follow up with physiotherapy, occupational therapy, rehabilitation, speech therapy & neuropsychiatry. 3. Discussion The availability of computed tomography (CT) scan as essential setup in ED enables physicians to readily diagnose brain injuries with high diagnostic accuracy. Once diagnosed, the site, size, course of injury, management plan are determined and also prognosis can be expected. Proper management of traumatic brain injury mainly depends on identification of primary injury and prevention of the secondary injury that essentially caused by hypoxia and hypotension [9–12]. Also, hypothermia, hypocapnia, hypoglycaemia and increased intracranial pressure are independent predictors of mortality [1,13]. Disability assessment generally uses the Glasgow Coma Scale (GCS) to stratify the risk into mild [13–15], moderate [9–12], and severe [3–8] head injury. Early initiation of therapy and the use of standardized guidelines in the management of moderate and severe TBI are essential to improve outcomes in pediatric patients [1,14]. Enteral access via gastrostomy tube is a common procedure in children. The laparoscopic approach to gastrostomy tube placement has been advocated by numerous centres. Several studies have compared percutaneous endoscopic gastrostomy (PEG) to laparoscopic gastrostomy (LG) placement in children finding the latter to have longer operative times but less serious intra- & post-operative complications [15,16]. With demonstration of safe early feeding after PEG, it seems to be a safe reasonable practice to initiate post-operative feeding as early as 4 h in patient undergoing laparoscopic gastrostomy with most patients tolerated with low risk for adverse events [16]. Laparoscopic gastrostomy is flourishing minimally invasive technique for gastrostomy tube placement in children. Based on advantages of the laparoscopic approach as direct visualization of the abdominal cavity, minimize unintentional hollow viscera injury as colon perforation, transfascial tacking suture to fix the tube, reduce risk of early tube dislodgement, & obviate the need to exchange tube for skin-level, balloon-based tube type, multiple studies have adopted for LG as the procedure of choice for children [16–21] as in our case. The gastrostomy was fixed to posterior rectus sheath and fascia by nonabsorbable sutures under vision. This method of tube fixation offers the least risk of tube dislodgement but increased surgical site infection rate [5,22]. Our patient was cared for by many other specialities as neurosurgery, rehabilitation, physiotheraspy, occupational therapy, speech, and pediatric neuropsychiatry as in Riachard et al., 2018 study [16]. The common complications of gastrostomy tube placement include surgical site infection, tube dislodgement, granulation tissue formation, peristomal leakage, stenosis, retraction, & proximal or distal tube migration [5]. Investigators have proposed different definitions and
Fig. 4. Two tiny mucosal pits remained and successfully cauterized by silver nitrate (green lines). (For interpretation of the references to color in this figure legend, the reader is referred to the Web version of this article).
Fig. 5. Schistosoma hematobium ova found in the excised granulation tissue.
Fig. 6. Schistosoma hematobium ova found in the excised granulation tissue. 3
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
categories for gastrostomy complications into either pre-procedural, intra-procedural or post-procedural, either early, or late, minor (approximately 23.6%) or major (about 8.8%), procedure-related, tube related, stoma-related, or related to patient feeding intolerance or change in clinical status as worsened gastroesophageal reflux disease (GERD) or aspiration pneumonia [5,21]. Granulation tissue has many nomenclatures as proposed by several authors. Some call it granulation tissue; others call it overgrowth of gastric mucosa or pseudotumour gastric mucosa as mentioned in the histopathology report [23]. The prevention and management of overgranulation around gastrostomy sites is a challenge for nurses to manage. Harris et al. [24] defines 3 types of overgranulation, Type one considers that overgranulation is caused by inflammatory factors that may be caused by an irritant at the wound edge. Type two is caused by moist environment. Type three is caused by cellular imbalance and has no specific treatment options other than to eradicate type 1 and 2 causation factors. Tenaka et al. [25] found that sprinkling salt reduced or removed gastrostomy overgranulation because salt draws water out of the edematous tissue, across the cell membrane, removing fluid and possible contaminants into the salt crystals [23]. Schiostsoma ova may be the causative factor (by chronic irritation) of granulation tissue formation. It reached the stomach via submucosal blood vessels from porto-mesenetric circulation or the mesentery if it is of Schistosoma mansoni type but in our case, no peristomal mesenteric hernia occurred and we hypothesize that the Schistosoma haematobium ova reached the stomach via retrograde manner from the urinary bladder retroperitoneal venous system or may be via still patent porto-systemic anastomosis. The disease control strategy for schistosomiais in Asia comprised three main approaches: (i) control of the snail intermediate host; (ii) treatment of all infected people; and (iii) concreting over the wetland habitat of the snail host. Existing control programs in endemic areas aimed at improving water supply, sanitation, toilet facilities, and hygiene such as healthy lifestyle & cultivation techniques have led to decreased infection rates [8]. Deworming is a preventive control measure but, to keep reinfection rates as low as possible, preventive education must be implemented widely and continuously throughout the region particularly in rural and remote communities in developing countries [8,26,27]. Schistosomiasis is transmitted by direct percutaneous penetration by cercariae, the free-swimming larval form of the worm, on exposure to infected fresh water snails. More than 200–240 million people world-wide are affected especially children in pre-school and school age groups, fishermen, and farmers. Commonly, these infections have subclinical symptoms in children and young adults [27]. Symptomatic infected cases presented firstly by maculpapular rash then after several weeks when the adult worms develop and start to lay eggs, the host defence reacts to the deposited ova leading to fever, headache, myalgia, hematuria, tender hepatosplenomegally, genitourinary tract disease, and bowel & hepatic fibrosis [28]. Anemia, nutritional deficiencies and stunted growth & cognitive delays are observed in chronic disease [29]. Diagnosis can be achieved by finding parasite ova in urine or stool and by serologic testing. On microscopy, the spike orientation helps in determination of species. Other supportive laboratory findings include eosinophilia, anemia, hypoalbuminemia [30]. The world health organization (WHO) aims at elimination of schistosomiasis globally by year 2025 [31]. The disease control program includes mass drug administration (MDA) or what is called preventive chemotherapy of praziquantel 40 mg/kg one dose in endemic areas given to high risk populations mainly preschool and school age children, adolescent girls, fishermen, farmers, and house-wife women. Also, improving access to drinking water, sanitation, hygiene, bovine treatment, snail control, modern irrigation & cultivation techniques, deworming strategies, and imple-
menting health education programmes with the community targeting the aforementioned high risk groups associated with annual MDA should be utilized in order to interrupt the worm life cycle and to achieve sustainable disease control [26,27]. 4. Conclusion Gastrostomy is one of the commonly performed procedures in children. It has several indications as swallowing disorders, failure to thrive, malnutrition, neurologic insult/impairment and various techniques including open, laparoscopic, endoscopic, or radiographic assisted. Schistosoma infestation can manifest in several ways but strangely the stomach. This mode of presentation should be considered to help diagnosis and to shed light spot on proper hygiene, infection prevention & control. Patient consent This report does not contain any personal information that could lead to the identification of the patient. Funding No funding or grant resources to disclose. Authorship All authors attest that they meet the current ICMJE criteria for Authorship. Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. List of abbreviations bpm CT ED E-FAST EMS EVD GCS GERD ICP ICU LG MBBS MDA MVC NGT PEG PICU RTA TAL TBI WHO
beat per minute Computerized tomography Emergency department Extended focussed assessment with sonography for trauma Emergency medical service External ventricular drainage Glasgow coma scale Gastroesophageal reflux disease Intracranial pressure Intensive care unit Laparoscopic gastrostomy Modified barium swallowing study Mass drug administration Motor vehicle collision Nasogastric tube Percutaneous endoscopic gastrostomy Pediatric intensive care unit Road traffic accident Tendon Achilles lengthening Traumatic brain injury World health organization
Appendix A. Supplementary data Supplementary data to this article can be found online at https:// doi.org/10.1016/j.epsc.2019.101377.
4
M.E. Mahmoud et al.
Journal of Pediatric Surgery Case Reports 53 (2020) 101377
References
[16] Hendrickson RJ, Poola AS, Sujka JA, Weaver KL, Rentea RM, Peter SDS, et al. Feeding advancement and simultaneous transition to discharge (FASTDischarge) after laparoscopic gastrostomy. J Pediatr Surg 2018;53:2326–30. [17] Zamakhshary M, Jamal M, Blair GK, Murphy JJ, Webber EM, Skarsgard ED. Laparoscopic vs percutaneous endoscopic gastrostomy tube insertion: a new pediatric gold standard?. J Pediatr Surg 2005;40(5):859–62. [18] Akay B, Capizzani TR, Lee AM, Drongowski RA, Geiger JD, Hirschl RB, et al. Gastrostomy tube placement in infants and children: is there a preferred technique?. J Pediatr Surg 2010;45:1147–52. [19] Richards MK, Li CI, Foti JL, Leu MG, Wahbeh GT, Shaw D, et al. Resource utilization after implementing a hospital-wide standardized feeding tube placement pathway. J Pediatr Surg 2016;51:1674–9. [20] Sunstrom R, Hamilton N, Fialkowski E, Lofberg K, McKee J, Sims T, et al. Minimizing variance in pediatric gastrostomy: does standardized perioperative feeding plan decrease cost and improve outcomes?. Am J Surg 2016;211:948–53. [21] McSweeney ME, Smithers CJ. Advances in pediatric gastrostomy placement. Gastrointest Endosc Clin N Am 2016;26(1):169–85. [22] Aprahamian CJ, Morgan TL, Harmon CM, Georgeson KE, Barnhart DC. U-stitch laparoscopic gastrostomy technique has a low rate of complications and allows primary button placement: experience with 461 pediatric procedures. Laparoendosc Adv Surg Tech A 2006;16:643–9. [23] Trautner E, Booth L. An audit around the use of salt in the treatment of overgranulation around PEG sites. Clin Nutr ESPEN 2018;28:273–4. [24] Harris A, Rolstad B. Hypergranulation tissue: a non-traumatic method of management. Ostomy Wound Manage 1994;40(5):20–30. [25] Tenaka H, Arai K, Fujino A. Treatment for hypergranulation at gastrostomy sites with sprinkling salt in pediatric patients. J Wound Care 2013;22(1):17–8. 20. [26] Yu W, Ross AG, Olveda RM, Harn DA, Li Y, Delia C, et al. Risk of human helminthiases: geospatial distribution and targeted Control. Int J Infect Dis 2017; 55:131–8. [27] Weatherhead JE, Hotez PJ, Mejia R. The global state of helminth control and elimination in children. Pediatr Clin N Am 2017;64:867–77. [28] Ross AG, Bartley PB, Sleigh AC, Olds GR, Li Y, Williams GM, et al. Schistosomiasis. N Engl J Med 2002;346(16):1212–20. [29] Hotez PJ, Fenwick A. Schistosomiasis in Africa: an emerging tragedy in our new global health decade. PLoS Neglected Trop Dis 2009;3(9):e485. [30] Gray DJ, Ross AG, Li YS, McManus DP. Diagnosis and management of schistosomiasis. BMJ 2011;342:d2651. [31] Hansen C, Paintsil E. Infectious diseases of poverty in children: a tale of two worlds. Pediatr Clin N Am 2016;63:37–66.
[1] Leetch AN, Wilson B. Pediatric major head injury not a minor problem. Emerg Med Clin N Am 2018;36:459–72. [2] Taylor CA. Traumatic brain injury–related emergency department visits, hospitalizations, and deaths—United States, 2007 and 2013. MMWR Surveill Summ 2017;66:1–16. [3] Pearce CB, Duncan HD. Enteral feeding. Nasogastric, nasojejunal, percutaneous endoscopic gastrostomy, or jejunostomy: its indications and limitations. Postgrad Med J 2002;78:198–204. [4] Pemberton J, Frankfurter C, Bailey K, Jones L, Walton JM. Gastrostomy matters−the impact of pediatric surgery on caregiver quality of life. J Pediatr Surg 2013;48:963–70. [5] Mason CA, Skarda DE, Bucher BT. Outcomes after laparoscopic gastrostomy suture techniques in children. J Surg Res 2018;232:26–32. [6] Utzinger J, Raso G, Brooker S, De Savigny D, Tanner M, Ornbjerg N, et al. Schistosomiasis and neglected tropical diseases: towards integrated and sustainable control and a word of caution. Parasitology 2009;136:1859–74. [7] Coulibaly JT, Panic G, Silué KD, Jana K, Jan H, Keiser J. Efficacy and safety of praziquantel in preschool-aged and school-aged children infected with Schistosoma mansoni: a randomised controlled, parallel-group, dose-ranging, phase 2 trial. Lancet Glob Health 2017;5:e688–98. [8] Ohta N, Waikagul J. Disease burden and epidemiology of soil-transmitted helminthiases and schistosomiasis in Asia: the Japanese perspective. J Parasit Trends 2006;23(1):30–5. [9] Adelson P. Guidelines for the acute medical management of severe traumatic brain injury in infants, children, and adolescents. Pediatr Crit Care Med 2003;4(3): 1–75. [10] McHugh GS, Engel DC, Butcher I, Steyerberg EW, Lu J, Mushkudiani N, et al. Prognostic value of secondary insults in traumatic brain injury: results from the IMPACT study. J Neurotrauma 2007;24(2):287–93. [11] Chang W-TW, Badjatia N. Neurotrauma. Emerg Med Clin N Am 2014;32(4):889– 905. [12] Spaite DW, Hu C, Bobrow BJ, Chikani V, Barnhart B, Gaither JB, et al. The effect of combined out-of-hospital hypotension and hypoxia on mortality in major traumatic brain injury. Ann Emerg Med 2017;69(1):62–72. [13] Wing R, James C. Pediatric head injury and concussion. Emerg Med Clin N Am 2013;31(3):653–75. [14] Blyth BJ, Bazarian JJ. Traumatic alterations in consciousness: traumatic brain injury. Emerg Med Clin N Am 2010;28(3):571–94. [15] Lantz M, Hultin Larsson H, Arnbjörnsson E. Literature review comparing laparoscopic and percutaneous endoscopic gastrostomies in a pediatric population. Int J Pediatr 2010;e507616.
5