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Journal of Indian Association of Pediatric Surgeons
     Journal of Indian Association of Pediatric Surgeons
Official journal of the Indian Association of Pediatric Surgeons         
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   Table of Contents - Current issue
Coverpage
October-December 2019
Volume 24 | Issue 4
Page Nos. 235-319

Online since Thursday, August 29, 2019

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EDITORIAL  

VISTAS of the future p. 235
Ashoke Kumar Basu
DOI:10.4103/jiaps.JIAPS_125_19  
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REVIEW ARTICLE Top

Tissue engineering and stem cell therapy in pediatric urology Highly accessed article p. 237
Shilpa Sharma, Devendra K Gupta
DOI:10.4103/jiaps.JIAPS_77_18  
The rapidly expanding field of tissue engineering along with stem cell therapy has a promising future in pediatric urological conditions. The initial struggle seemed difficult in renal regeneration but a functional biounit has been developed. Urine excretion has been demonstrated successfully from stem cell-generated embryonic kidneys. Three-dimensional (3D) stem cell-derived organoids are the new paradigm in research. Techniques to regenerate bladder tissue have reached the clinic, and the urethra is close behind. 3D bioprinted urethras would soon be available. Artificial germ cells produced from mouse pluripotent stem cells have been shown to give rise to live progeny. Myoblast and fibroblast therapy has been safely and effectively used for urinary incontinence. Stress urinary incontinence has been clinically treated with muscle-derived stem cells. Skeletal muscle-derived stem cells have been shown to get converted into smooth muscle cells when implanted into the corpora cavernosa in animal models. This review encompasses the various experimental and clinical developments in this field that can benefit pediatric urological conditions with the contemporary developments in the field.
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ORIGINAL ARTICLES Top

Can grafted tubularized incised plate urethroplasty be used to repair narrow urethral plate hypospadias? Its functional evaluation using uroflowmetry p. 247
Pradyumna Pan
DOI:10.4103/jiaps.JIAPS_151_18  
Aim: Using uroflowmetry, the aim of this study is to determine the functional results of the grafted tubularized incised plate (GTIP) urethroplasty used to repair poor urethral plate hypospadias. Settings and Design: Seventy-one patients (mean age: 5.7 years, follow-up: 1–5.5 years) were selected from those who underwent surgery using the GTIP technique from 2013 to 2015 at our institution. Methods: Patients included were able to void voluntarily and had no fistula. The flow pattern, maximum urinary flow rate (Qmax), voided volume (vv), average flow rate, and voiding time were measured. The results were expressed as percentiles and interpreted according to Siroky nomogram. The Qmaxwas considered normal if >25th percentile, as equivocally obstructed when in the 5th–25th percentile, and obstructed if <5th percentile. Results: Hypospadias was distal in 45, mid penile in 17 and proximal penile in 9. The uroflow curve was bell-shaped in 24 (30%), interrupted in 9 (14%), slightly flattened in 31 (46%), and a plateau in 7 (10%). Flow rate nomograms revealed that 49 (68%) were above the 25th percentile, 9 (17%) were below the 5th percentile, and 13 (15%) were between these ranges. Eleven patients showed improvement in the flow curve and maximum urinary flow rate (Qmax) in follow-up uroflowmetry. Conclusion: GTIP repair provides satisfactory functional results. A long-term follow-up is needed to confirm these results.
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Surgical correction of pectus excavatum using a rib graft strut following excision of costal cartilages p. 252
Veereshwar Bhatnagar, Nitin Sharma, Anjan Dhua, Manisha Jana
DOI:10.4103/jiaps.JIAPS_68_18  
Background: A number of techniques are described for correction of pectus excavatum (PE). This article describes the experience with an innovative procedure which combines features from the Ravitch and Nuss procedures without using prosthetic material. Methods: This cross-sectional study included 12 cases of PE from January 2000 to March 2017 managed by excision of deformed costal cartilages and support to the thoracic cage using an autologous free rib graft as a strut. Indication for surgery was Haller's Index above 3.2 with or without respiratory distress. Noncontrast computed tomography scans were done at 6 months after surgery to document the position of the strut and to see the final correction and new Haller's Index, respectively. Results: The male-to-female ratio was 2:1. Preoperative Haller's Index in all cases was >3.2 (range 3.25–14). The average age at surgery was 5 years and 8 months (range: 7 months–15 years). Mean duration of hospital stay was 11 days (range 5–16 days).The 11th rib was used commonly although in two cases, the 10th rib was used as the 11th rib was considered relatively short. Pericardial effusion requiring strut removal was seen in one case; in another case, removal of the rib was needed because of nonhealing of a delayed dehisced surgical wound. Others had an uneventful postoperative period. The mean postoperative Haller's Index was 2.75 (range 2.0–7). Conclusion: This modified procedure using an autologous rib strut is technically feasible and reproducible even with limited facilities and gives excellent results.
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Outcome of care provided in neonatal surgery intensive care unit of a public sector tertiary care teaching hospital of India p. 257
Vijaydeep Siddharth, Shakti Kumar Gupta, Sandeep Agarwala, Sidhartha Satpathy, Prabudh Goel
DOI:10.4103/jiaps.JIAPS_177_18  
Aims: There is limited literature on the outcome of care in intensive care units (ICUs), especially when it comes to neonatal surgical units. Hence, this study was aimed to observe the outcome of care provided in the neonatal surgery ICU (NSICU) at an apex tertiary care teaching institute of North India. Methods: A descriptive, observational study was carried out through retrospective medical record analysis of all the patients admitted in NSICU from January to June 2011. Results: In NSICU, from January to June 2011, 85 patients were admitted. More than two-third (69.9%) patients were admitted through the emergency department. Of the total admitted patients, 69.9% were male. Mean and median age of the admitted patients were 6.31 and 2 days (range 0–153 days), respectively. The most common diagnosis was esophageal atresia with tracheoesophageal fistula (36.1%).Within a day of admission at NSICU, 88% patients underwent surgical intervention. Of the total admitted patients, 56.6% required mechanical ventilation with 3.57 days (range 0–31 days) of mean duration of mechanical ventilation. Reintubation rate (within 48 h of extubation) was observed to be 15.7%, and 27.7% (23) of the patients required vasopressor support during their NSICU stay. Patients who developed postoperative complications were 34.25%, with the most common being wound infection/discharge/dehiscence. Two patients were readmitted within 72 h of their discharge/transfer out from the NSICU. Conclusion: NSICU survival rate was 85.5% and net death rate was observed to be 14.5%. Sepsis was the major reason for mortality in NSICU.
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Pediatric thoracoabdominal trauma: Experience from a tertiary care center p. 264
Vijay Kumar Kundal, Pinaki Ranjan Debnath, Atul Kumar Meena, Shalu Shah, Prafull Kumar, Shyam Sunder Sahu, Amita Sen
DOI:10.4103/jiaps.JIAPS_152_18  
Aim: The aim of this study is to assess the pattern and mode of thoracoabdominal trauma and anatomical organ involvement, type of management employed, and overall outcomes in the pediatric population. Materials and Methods: A retrospective study conducted at a tertiary hospital over a period of 38 months with a total of 198 pediatric patients <12 years of age with a history of abdominal and chest trauma between July 2014 and September 2017 were reviewed. Case files of all pediatric patients were evaluated. Information regarding age, sex, mechanism of injury, site of injury, mode of injury, nature of the injury, definitive treatment required, whether conservative or surgical and outcome of patients was evaluated. site of injury, mode of injury, nature of the injury, definitive treatment required, whether conservative or surgical and outcome of patients was evaluated. Results: A total of 198 patients of thoracoabdominal trauma patients were studied. The majority of pediatric thoracoabdominal trauma cases were observed in males (n = 128, 64.64%) and females comprise only 35.35% (n = 70). Fall was the most common mode of trauma (58.08%) followed by road traffic accidents (30.30%), thermal injuries (9.09%) assaults in order of decreasing trends. The abdomen was the most common anatomical site of the injury (45.95%) followed by combined thoracoabdominal trauma (32.32%) followed by thoracic trauma (21.71%). Among the thoracic trauma, the most common comprised the lung contusions (37.20%) followed by the lung laceration comprising (25.58%) and rib fractures comprised only 20.93%. Among the abdominal trauma, the most common organ of injury was the liver (36.26%) followed by splenic trauma in 19.78% of patients. Approximately, 85% of patients were managed conservatively, and only 15% required major surgical intervention in the form of laparotomy and repair of bowel perforation, thoracotomy and ligation of bleeding intercostal vessels, partial and total splenectomy, repair of the liver laceration, and nephrectomy for the patient of Grade 5 renal injury with expanding retroperitoneal hematoma. Three patients died, one of which was Grade 5 renal injury with expanding retroperitoneal hematoma, two others were cases of combined thoracoabdominal trauma with massive hemothorax and both liver and splenic injury. Conclusion: The study defines the pattern of thoracoabdominal trauma, mode of trauma, and the prevalence of different organs involved in both the chest and abdominal cavity. We concluded that main indications for performing an operative intervention included severe hemodynamic instability, pneumoperitoneum, massive pneumohemothorax with significant shift and definitive confirmation of oral contrast leak on computed tomography films. A detailed trauma registry in our set up seems important for managing pediatric thoracoabdominal trauma.
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Age is not a criterion in patient selection for kasai portoenterostomy p. 271
Priya Ramachandran, Mohamed Safwan, Vidya Tamizhvanan, Muthukrishnan Saravana Balaji, Ashitha K Unny, Mukul Vij, Mohamed Rela
DOI:10.4103/jiaps.JIAPS_182_18  
Aims: The aim of our study was to compare the outcome of Kasai portoenterostomy (KPE) in children with biliary atresia (BA) older than 90 days to children less than 90 days and to study its safety and efficacy in children older than 90 days. Subjects and Methods: Relevant data were collected from our prospectively maintained database of all children with BA who underwent KPE over a 5-year period. Children were divided into two groups: Group 1 ≤90 days and Group 2 >90 days. Data analyzed and compared included total and direct bilirubin, aspartate aminotransferase-to-platelet ratio index (APRI), and the outcome of procedure which was defined as a serum direct bilirubin <2 mg/dl within 6 months after surgery. Standard statistical tests were used for analysis. Results: Out of 62 children, 45 children were in Group 1 and 17 children were in Group 2. Children in Group 2 had similar total and direct bilirubin compared to children in Group 1. APRI, an indicator of fibrosis, was significantly increased in Group 2 (P = 0.08). About 47% of children in Group 2 had Stage III fibrosis on liver histology compared to 22% of children in Group 1. None of the children in Group 2 had synthetic liver failure (refractory ascites, hypoalbuminemia, or coagulopathy unresponsive to Vitamin K) or portal hypertension. KPE was successful in 29.4% of children in Group 2 and 44% in children in Group 1. There was no perioperative mortality in our group. Conclusions: KPE was successful in a third of children over 90 days of age and can be safely performed in this group. In the absence of synthetic liver failure, age should not be a disqualification for performing KPE.
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Urethral duplication in children: Experience of twenty cases p. 275
Sajad Ahmad Wani, Narendra Babu Munianjana, Viney Jadhav, S Ramesh, BC Gowrishankar, J Deepak
DOI:10.4103/jiaps.JIAPS_164_18  
Background and Objective: Urethral duplication is a rare congenital anomaly having multiple anatomical variants and varied presentations. Multiple surgical techniques have been described for this anomaly, and no surgical technique is ideal. The aim of this study was to present our experience of twenty cases of urethral duplication in the management of this anomaly. Materials and Methods: Retrospectively over a period of 10 years from 2006 to 2016, records of all urethral duplications were reviewed. All available records were evaluated for clinical presentation, imaging studies (micturating cystourethrogram, retrograde urethrogram, ultrasonography, and cystourethroscopy), and classified according to the Effmann's classification. The intraoperative details, outcome, and follow-up (including cystoscopy and contrast studies) were noted. Results: There were 20 patients of urethral duplications. Age of patients ranged from 3 months to 9 years with a mean age of 4.6 years. The details of management, outcome, and follow-up were obtained. Conclusion: Management varies with the symptoms and the severity of the anomaly. Y-type urethral duplication is more complex and challenging and requires extensive urethral reconstruction. Combined use of the bladder and buccal mucosa for reconstruction gives better results than bladder mucosa alone.
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Prediction of gap length by plain radiograph of chest with nasogastric tube in the upper esophagus in patients with esophageal atresia and distal tracheoesophageal fistula p. 281
Muffazzal Rassiwala, Partap Singh Yadav, Subhasis Roy Choudhury, Niyaz Ahmed Khan, Shalu Shah, Pinaki Ranjan Debnath, Rajiv Chadha
DOI:10.4103/jiaps.JIAPS_184_18  
Aim: This study was aimed at prediction of the gap length between the two esophageal ends in cases of esophageal atresia and distal tracheoesophageal fistula (EA-TEF) by preoperative radiography with a nasogastric tube in the upper esophagus and its correlation with intraoperatively measured gap length. Materials and Methods: All consecutive cases of EA-TEF were prospectively included in this study. Plain radiographs were taken with an 8 Fr nasogastric tube inserted in the upper esophageal pouch till its arrest. The patients were grouped into T1-T2; T2-T3; T3-T4; and T4 groups depending on the thoracic vertebral level of arrest of the NG tube on the radiograph. Intraoperative gap between the two esophageal ends was measured with Vernier caliper, and the patients were grouped into A, B, and C groups based on gap length (gap length >2.1 cm; >1–≤2 cm; and ≤1 cm). The operative gap groups were compared with the radiography groups. Results: A total number of 118 cases were included over a period of 3 years. The arrest of nasogastric tube at T1-T2 and T2-T3 vertebral level corresponded to gap length Group A in 39/41 (95.12%) * patients. In gap length Group B, the arrest of tube at T2-T3 and T3-T4 vertebral level was seen in 44/44 (100%) * patients, in gap length Group C, the arrest of tube was noted at T3-T4 and T4 vertebral level in 31/33 (93.93%) * patients (*P < 0.001). Conclusion: Prediction of gap length by vertebral level of arrest of the nasogastric tube in the upper pouch in a preoperative chest X-ray correlated well with intra operatively measured gap length in cases of EA-TEF.
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CASE REPORTS Top

Congenital segmental dilatation of jejunum: A rare entity p. 285
Amar A Shah, Anirudh V Shah
DOI:10.4103/jiaps.JIAPS_201_18  
Segmental dilatation of the intestine is a rare disease and mostly involves the ileal segment. It commonly presents in the neonatal period and early infancy with symptoms of partial or total obstruction. We report a rare case of an isolated jejunal segmental dilatation in a 7-year-old girl. The child presented with malnutrition and signs of subacute obstruction. The diagnosis was confirmed intraoperatively, and the dilated segment was resected. Although the etiology remains unknown, we discuss its clinical aspects and relevant literature.
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Transverse testicular ectopia with fused vas deferens: A case series p. 288
Sivasankar Jayakumar, Meena Agrawal
DOI:10.4103/jiaps.JIAPS_156_18  
Transverse testicular ectopia (TTE) is a rare anomaly where both gonads descend towards the same side of the hemiscrotum. Although more than 100 cases of TTE are reported, TTE with fused vas are extremely rare. We present our series of 4 TTE patients with fused vas [4, 7, 9 and 26 months old]. Three cases were diagnosed at the time of inguinal exploration and one during a planned laparoscopic inguinal hernia repair. An indirect inguinal hernia with TTE was noted with a fused proximal vas deferens in all 4 cases [Figure 1]. Inguinal herniotomy, ipsilateral orchidopexy along with contralateral transeptal orchidopexy (testis brought down through the inguinal canal on the same side of hernia) [Figure 2] was performed in all patients without any complication. None of the patients had uterine tissue. Testicular biopsies, Array and karyotyping (46XY) were normal in all 4 patients. Follow-up ultrasound doppler scans revealed normal testes with good vascularity in all patients. Fused vas with TTE is rare but transeptal orchidopexy is feasible with excellent outcome.
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Biliary pseudocyst: A rare complication of antenatal perforation of forme fruste choledochal cyst p. 291
Mrinal Arora, Jaswinder Kaur, Satish Kumar Aggarwal
DOI:10.4103/jiaps.JIAPS_212_18  
Spontaneous perforation of choledochal cyst (CDC) is rare with an incidence of 1.8%–7%. Perforation with pseudocyst formation is even rarer, there being only two case reports. We report the first case of antenatal perforation of CDC with biliary pseudocyst formation in a newborn and its management.
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Previously undescribed anomalies of hepatic artery and portal venous anatomy in a case of extrahepatic biliary atresia and its implications p. 294
Prabudh Goel, Minu Bajpai, Kanika Sharma, Priyanka Naranje
DOI:10.4103/jiaps.JIAPS_132_18  
A search on PubMed and Web of Science revealed scarcity of the literature on anomalies of hepatic artery or portal vein and the presence of arterioportal fistula in biliary atresia; although, it has long-lasting implications for both the patient and the surgeon, including hepato-pancreato-biliary surgeons, pediatric surgeons (who perform Kasai's portoenterostomy), liver transplant surgeons, and interventional radiologists. We report a case of extrahepatic biliary atresia with multiple anomalies involving the hepatic arteries, portal vein, cystic artery, arterioportal fistula and shunting, intrahepatic portal vein radicals, kidney, and external genitalia. The merits of the case from various standpoints including its implications for etiopathogenesis, caution during surgical anesthesia or postoperative management, and enrichment of the literature have been discussed.
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Thoracoscopic left cardiac sympathetic denervation in a child with refractory long QT syndrome p. 297
Sachit Anand, Vishesh Jain, Sandeep Agarwala, Sakshi Sachdeva, Shyam Sunder Kothari
DOI:10.4103/jiaps.JIAPS_144_18  
Long QT syndrome is a cardiac disorder which presents with recurrent syncopal attacks and has risk of sudden cardiac death. A 5-year-old boy presented to us with this syndrome. The child was symptomatic despite medical management and was successfully managed with cardiac denervation. The current report highlights the efficacy and safety of the use of video-assisted thoracoscopic surgery for this procedure.
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Congenital dermal facial temporal sinus: Rare presentation of intraorbital dermoid in children p. 300
Nandita A Saxena, Akshay G Nair, Bharati K Kulkarni, Pooja M Multani, Hemant N Lahoti, Shyam S Borwankar, Dipesh Goel
DOI:10.4103/jiaps.JIAPS_153_18  
We present a rare case of an intraorbital dermoid which was associated with a small temporal region dermal sinus in a 3-year-old child. This got infected and the child presented with orbital cellulitis. Definitive surgery involved excision of all the dermal elements using a superficial and intraorbital approach. We stress the need to evaluate, apparently benign lateral facial dermal sinuses as they may be the pointers of the underlying pathological deep dermoid cysts to avoid complications.
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Total intestinal atresia: Revisiting the pathogenesis of congenital atresias p. 303
Neel Aggerwal, Nidhi Sugandhi, Harshita Kour, Goutam Chakraborty, Samir Kant Acharya, Amit Jadhav, Deepak Bagga
DOI:10.4103/jiaps.JIAPS_204_18  
Despite various theories to explain the pathogenesis of atresias, the exact mechanism is still controversial. Currently, atresias are believed to result from vascular accidents and less likely due to the failure of recanalization. We report a case which challenges this belief. A 1-day-old neonate was explored for suspected jejunal atresia. Apart from Type III jejunal atresia, 15 cm from DJ junction, there was surprisingly no distal lumen in the intestine from jejunum till rectum. Multiple enterotomies revealed the whole of the remaining jejunum, ileum, and large colon to be a solid cord-like structure. No distal luminal contents or histopathological evidence of ischemic damage was seen, thus suggesting the probable etiology to be a failure of recanalization of the gut cord rather than a late vascular accident. Such rare cases provide insights into possible embryogenetic mechanisms which can then aid in formulating preventive measures.
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Cervical giant immature teratoma in a newborn: A challenge for survival p. 307
Nagarjuna Kumbha, Allu Rohita, Sarweswar Porca Reddy, Ambati Karuna Sagar
DOI:10.4103/jiaps.JIAPS_136_18  
Teratomas are a conglomerate of heterogenous cells arising from totipotent germ cells. Cervical teratomas occur 1 per 20,000 to 40,000 live births constituting 1.6-9.3% of all paediatric teratomas . They can present antenatally with cervical mass or polyhydramnios and postnatally with respiratory distress and facial disfigurement. Complete excision of the tumour prevents malignant transformation. Timing of the surgery is based on severity of airway compromise. Surgical outcome and survival depend on pre-existing pressure effects, operative injuries to the vital structures and also co-existing comorbidities. One such complicated case of giant cervical teratoma is described here.
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IMAGES Top

Accessory scrotum p. 309
Ritika Parmar, Meera Luthra, Dinesh Raj
DOI:10.4103/jiaps.JIAPS_181_18  
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LETTER TO THE EDITOR Top

Ileoileocolic intussusception secondary to isolated ectopic pancreatic tissue: An uncommon case Highly accessed article p. 310
Vipul D Yagnik, Sushil Dawka, Sunil Prajapati, Bhavna Mehta
DOI:10.4103/jiaps.JIAPS_245_18  
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Etiopathogenesis of chordee in hypospadias p. 311
Sachchidananda Das, Sudarsan Sen
DOI:10.4103/jiaps.JIAPS_91_19  
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Duodenal teratoma: A rare diagnostic and therapeutic challenge p. 313
Archana Puri, Arun Chauhan, Sunita Bhalla
DOI:10.4103/jiaps.JIAPS_226_18  
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Basal cell carcinoma with xeroderma pigmentosum in an 8-Year-old girl p. 314
Aditya Pratap Singh, Maryem Ansari, Arvind Kumar Shukla
DOI:10.4103/jiaps.JIAPS_162_18  
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Dhaga syndrome: A perplexing dilemma of “What Lies Beneath” p. 317
Saurabh Shyam Garge, Geetika Paliwal, Bhupendra Lodwal
DOI:10.4103/jiaps.JIAPS_228_18  
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BOOK REVIEW Top

Pediatric surgery simplified – Dr. Minakshi Bhosale: A book review p. 319
Ramesh Santhanakrishnan
DOI:10.4103/jiaps.JIAPS_70_19  
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