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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
July-September 2018
Volume 23 | Issue 3
Page Nos. 111-177

Online since Wednesday, July 4, 2018

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Biliary atresia, changing trends in management: Outlook of a pediatric liver transplant surgeon Highly accessed article p. 111
T Renu Kumar
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Spontaneous pneumoperitoneum in pediatric patients: Dilemmas in Management Highly accessed article p. 115
Rahul Gupta
Context: Pneumoperitoneum is typically a surgical emergency, particularly in neonates. However, pneumoperitoneum can rarely present without gastrointestinal perforation which is known as spontaneous pneumoperitoneum. Aims: This study aims to analyze the outcome of pediatric patients with spontaneous pneumoperitoneum and describe our experience. Settings and Design: A retrospective study performed from January 2014 to May 2017. Materials and Methods: The study included all pediatric patients admitted in a tertiary care institute. Results: There were 9 patients with 5 males and 4 females, out of which 7 were neonates. At presentation, respiratory distress was a common complaint (8/9). 5 out of all patients developed abdominal distension. Associated malformations were seen in 5 patients. Pneumoperitoneum was preceded by mechanical ventilation in 5 patients. Abdominal examination revealed soft distension with relaxed abdominal wall in 7, while marked distension was present in 2 cases. Features suggestive of peritonitis were absent in all the patients. Abdominal radiographs revealed free air in all, while, radiographic evidence of pneumothorax was seen in 2 patients. Conservative management without any operative intervention was performed in 3 patients with favorable outcome. Laparotomy was performed in 1 patient; negative for any perforation and outcome was unfavorable. In 5 patients, abdominocentesis was performed followed by abdominal drain placement. No patient had any evidence of leakage of intestinal contents from gastrointestinal tract after insertion of abdominal drain. Favorable outcome was present in 5 patients. Out of 4 cases with unfavorable outcome, 3 patients were having esophageal atresia. Conclusions: A high index of suspicion for spontaneous pneumoperitoneum must be present when dealing with pediatric patients, especially neonates with pneumoperitoneum and without clinical signs of peritonitis. A comprehensive clinical evaluation with sepsis screen must be carried out to supplement the diagnosis. Pneumoperitoneum in pediatric patients, especially neonates, is not always an absolute indication for surgery.
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Pelviureteric junction obstruction with crossing lower polar vessel: Indicators of preoperative diagnosis p. 123
Alisha Gupta, Anjan Dhua, Sandeep Agarwala, Veereshwar Bhatnagar
Introduction: A crossing lower polar vessel (CLPV) is found in 11%–20% of children of pelviureteric junction obstruction (PUJO). Preoperative imaging (Doppler or magnetic resonance angiography (MRA)) may help but does not form a part of routine diagnostic workup. An attempt has been made here to evaluate clinical and imaging features (ultrasound and renal dynamic scan [RDS]) in children of PUJO with CLPV and define variables that could provide a diagnostic clue to its presence. Materials and Methods: Records of children having PUJO with CLPV over 10 years (2006–2015) were reviewed retrospectively. Their demographic profile, clinical presentation, imaging features, management, and outcomes were evaluated. Results: Of 372 children with PUJO, 21 (5.6%) had a CLPV. Median age at presentation was 7 years (range 4 months–11 years). Presenting features included pain (66.6%), urinary tract infection (14.3%), antenatally diagnosed fetal hydronephrosis (14.3%) and lump (4.8%). All had an obstructive drainage on RDS and mean split renal function (SRF) of the affected kidney was 32.5% ± 15.65%. The majority had mild-moderate hydronephrosis with intrarenal pelvis. None of the ultrasound images suggested the presence of a CLPV. Diagnosis of a CLPV was made intraoperatively in all. Dismembered pyeloplasty anterior to the vessel was done in the majority (80.9%). Mean SRF on postoperative RDS improved to 36.6% ±17.76% with nonobstructive drainage. All were asymptomatic at a mean follow-up of 34.5 ± 17.5 months (range 2–56 months). Conclusions: Pain is the predominant presenting feature in these patients who present at an older age. Despite older age at presentation, these patients have well-preserved renal function and mild hydronephrosis. No specific RDS or ultrasound findings can predict the presence of a CLPV. As the incidence of CLPV is <10% and management is essentially same as PUJO with intrinsic obstruction, preoperative Doppler or MRA are extraneous.
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Positioning the umbilicus in african newborns with large anterior abdominal wall defects p. 127
Olakayode Olaolu Ogundoyin, Dare Isaac Olulana, Taiwo Akeem Lawal
Objective: We tried to determine the normal position of the umbilicus in African newborns with a potential application in aesthetically acceptable umbilical reconstruction. Subjects and Methods: The study involves a cross-sectional study of 896 healthy neonates who underwent clinical abdominal examination and measurement of the distances between the xiphoid process and the umbilicus (XU), xiphoid process and the pubis (XP), umbilicus and the pubis (UP), umbilicus and the anterior superior iliac spine (UASIS), abdominal girth (AG), and inter anterior superior iliac spines (IASIS) distance. The gestational age at delivery, age at measurement, sex, AG measured across the umbilicus, birth weight (BW), body length, and body mass index of the newborns were recorded. The measurements were used to determine the location of the umbilicus and Pearson's correlation analysis performed to determine the relationships of the recorded neonatal parameters with the measurements. Results: The mean XP was 11.97 ± 2.09 cm, XU = 7.94 ± 2.74 cm, UP = 4.26 ± 1.21 cm, UASIS = 5.79 ± 1.04 cm, and IASIS = 10.25 ± 1.54 cm. The BW, length, and AG significantly correlated with the distance between the XU, umbilicus and pubis, umbilicus and anterior superior iliac spine as well as the distance between the two anterior superior iliac spines. Conclusion: The position of the umbilicus is dependent on the selected participants' characteristics. We suggest that a UP: XU ratio of 0.55 should be used to position the umbilicus during umbilical reconstruction in African neonates.
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A clinical study on congenital diaphragmatic hernia in neonates: Our institutional experience p. 131
Jayalaxmi Shripati Aihole, Aruna Gowdra, Deepak Javaregowda, Vinay Jadhav, M Narendra Babu, Ravidra Sahadev
Background: Congenital diaphragmatic hernia (CDH) is a complex developmental defect having a multifactorial etiology; i majority of cases (~80%), the cause is not known. Survival rates for patients with CDH have increased over the past decade with early prenatal detection and better postnatal management including surgery. Clinical profile and the outcome of 83 CDH neonates were studied and analyzed over a period of 12 years in our institute. Aims and Objectives: The clinical study was to analyze the clinical profile and outcome of CDH among the neonates in a tertiary care referral neonatal and pediatric center in Karnataka, India. Materials and Methods: This was a retrospective and prospective observational study conducted from January 2005 to March 2017, over a period of 12 years in a tertiary care referral neonatal and pediatric center in southern India. Clinical characteristics and risk factors of 83 neonates admitted and diagnosed with CDH were compared between survivors and nonsurvivors both preoperatively and postoperatively. Neonates with clinical and intraoperative diagnosis of diaphragmatic eventration were not included in this study. Multivariate logistic regression analysis was performed to determine independent predictors for mortality. Results: A total of 83 neonates admitted and diagnosed with CDH were included in this study; 73 of them underwent surgical repair. The total survival rate in neonates with CDH was 70/83 (84.33%) and the overall operative mortality was 3/73 (4.1%). There was a significant difference between CDH neonates who survived 70/83 (84.33%) and those who died 13/83 (15.67%), in the age on admission, 5 min Apgar score, onset of respiratory distress, preoperative ventilation, the presence of persistent pulmonary hypertension of the newborn (PPHN), high-frequency oscillatory ventilation (HFOV), and length of hospital stay with P < 0.05. Using multivariate logistic regression analysis, the following factors independently predicted mortality: onset of respiratory distress in hours (odds ratio: 0.5, 95% confidence interval: 0.37–0.82) and preoperative ventilation (odds ratio: 0.02; 95% confidence interval: 0.0028–0.1558). When we compared CDH neonates who survived after surgery (n = 70) with those who expired (n = 3) postoperatively, there was a significant difference in the gestational age in weeks, side of CDH, PPHN, HFOV and length of hospital stay with P < 0.05. Conclusion: CDHs are common on the left side with fairly good prognosis. Though, the right-sided CDH are rare; they do carry a good prognosis, as it was seen in our experience.
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The modified multilayer coverage of urethroplasty for distal hypospadias p. 140
Mohamed Oulad Saiad
Purpose: Our purpose is to present the modified multilayer coverage to prevent fistula and to present also the glans dissection respecting the continuity between glans and corpus spongiosum. We think an important factor for glans vascularization that prevents glans disruption for patients with distal hypospadias and report the follow-up. Introduction: Fistula and glans disruption are still the most frequent complications of hypospadias surgery. Neourethral coverage is a mandatory step in this challenging surgery; it reduces the rate of fistula. Materials and Methods: Between September 2013 and December 2015, the modified multilayer coverage using two or three intermediate layers was performed in 110 patients with distal and midshaft hypospadias. Follow-up based on local examination, visual assessment of urine stream, and parent satisfaction was recorded at 1, 3, 6 months, 1 year, and yearly thereafter. Patients lost for the follow-up and the circumcised patients with a bad quality remaining dorsal dartos are excluded from the study. Results: Intraoperatively, the decision whether two or three intermediate layers should be performed depending on the type of the hypospadias and the presence of hypoplastic urethra. Among the 88 patients retained for the study, 86 patients presented satisfactory results, but one patient had a fistula that healed spontaneously and the second patient had a glans disruption. Conclusions: The modified multilayer coverage with a wings glans dissection respecting the continuity of spongiosum and glans is a reliable procedure to reduce the rate of fistula and glans disruption in anterior and midshaft hypospadias.
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Comparison of anatomical landmarks and dimensions in a hypospadiac glans with those of a normal glans p. 144
Anjan Kumar Dhua, Sachit Anand, Sandeep Agarwala, Veereshwar Bhatnagar
Aim of the Study: The aim of this study is to establish ventral glans length (VGL), meatus (M) size, and their ratio VGL/M (R) in normal Indian boys and to compare these with the respective equivalent dimensions in boys with distal and mid-penile hypospadias using anatomic landmarks. Methods: Normal boys were designated as Group A (n = 108), and the boys with hypospadias were designated as Group B (n = 81). The anatomical landmarks marked on the glans were measured using a digital camera and appropriate software. Results: The mean age of the boys in Groups A and B was 4.26 ± 3.59 (range 0.5–12) and 3.82 ± 2.85 (0.7–11) years, respectively. The observed mean values in Group A for VGL-A, M-A, and R-A were 3.05 ± 1.27 (range 1.19–8.09), 4.3 ± 1.23 (range 1.61–7.04), and 0.8 ± 0.45 (range 0.27–2.1) mm, respectively. The observed mean values in Group B for VGL-B, M-B, and R-B were 3.77 ± 0.81 (range 12.41–5.2), 3.27 ± 0.71 (range 2.25–5.34), and 1.17 ± 0.26 (range 0.84–1.86) mm, respectively. Comparison of R-A and R-B showed a statistically significant difference (P < 0.0001). Conclusions: The dimensions and the studied ratio between a hypospadiac glans and a normal glans differed significantly. The ratio between VGL and M was 0.8 in normal boys. In comparison, the ratio between the equivalent VGL and M in the hypospadiac boys was 1.17. During glansplasty in surgery for hypospadias, this factor should be taken into consideration to avoid the creation of long and tight ventral glans closure.
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Revision surgery in the management of anorectal malformations: Experience from a tertiary center of India p. 148
Basant Kumar, Vijai Dutta Upadhyaya, Manish Kumar Gupta, Srinivasa Kishore, JB Nijagal Mutt, Rajanikant Yadav, Sheo Kumar
Aim and Objectives: Despite the significant advancements in the management of anorectal malformations (ARMs), there are various surgical and functional complications reported. Complications are closely related with the surgical techniques adopted and the types of malformations. In this article, we present our experiences with ARM patients who required reoperation after unsuccessful previous surgeries or who had developed complications related to the previous surgical techniques. Materials and Methods: We retrospectively reviewed clinical and electronic records of all the patients with ARM who were operated for ARMs in our institute from June 2010 to May 2016. All ARM patients who needed reoperation were included in the study. These patients were previously operated outside our institute and referred to us with ongoing problems of constipation, stool impaction with overflow incontinence, perineal soiling, and difficult urination. Results: There were 31 patients (M:F = 2.1:1) of ARM, reoperated for 38 indications during the above-mentioned period. Five patients had more than one problem. Presentation included neoanal stenosis (11), complete obliteration of neoanus (2), malpositioned neoanus (2), persistent/recurrent rectourethral fistula (3), iatrogenic rectovaginal fistula (4), rectal prolapse (5), large widened neoanus with soiling (2), and urethral stricture (2), which required revision interventions. Six patients had megarectum. All patients showed improvement in their symptoms after revision surgery, but 10 (41.7%) patients required further regular bowel management program (BMP) to avoid the soiling and constipation. Fourteen (58.3%) patients stayed clean without regular BMP. Conclusion: All these complications had clear explanations and are well described in the literature. Revision surgery in such patients had fair outcome, but some sort of BMP was required. Both posterior sagittal anorectoplasty and anterior sagittal anorectoplasty are excellent techniques for revision surgery with few simple modifications.
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Chest wall fibromatosis with congenital muscular torticollis: Two forms of deep fibromatosis in a child p. 153
Sachit Anand, Anjan K Dhua, Veereshwar Bhatnagar, Devasenathipathy Kandasamy, Sudheer Arava
Desmoid tumors (fibromatosis) are intermediate grade, locally aggressive soft-tissue tumors. A rare case of chest wall fibromatosis in a girl with congenital muscular torticollis is being reported. This report also highlights the need for long-term surveillance in such cases, despite being a benign pathology.
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Littoral cell angioma: A rare cause of pediatric thrombocytopenia p. 156
Mitul Jasani, Anirudh Shah, Amar Shah
Littoral cell angioma is a rare primary splenic vascular tumor, originating from the littoral cells lining the red pulp sinuses of the spleen, with variable presentation. Here, we present the case of a 2-year-old male child presenting with chronic thrombocytopenia which resolved after splenectomy.
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Posterior reversible encephalopathy syndrome after augmentation cystoplasty in a child with neurogenic bladder p. 158
Mudasir Ahmad Magray, Gowhar Nazir Mufti, Nisar Ahmad Bhat, Aejaz Ahsan Baba, Mudasir Hamid Buch, Faheem Ul Hasan, Shahid Bashir Banday
Posterior reversible encephalopathy syndrome (PRES) or leukoencephalopathy syndrome was introduced into clinical practice in 1996 by Hinchey et al., to describe unique syndrome, clinically expressed during hypertensive and uremic encephalopathy, eclampsia, and immunosuppressive therapy. Hyperperfusion with resultant disruption of the blood–brain barrier results in vasogenic edema, but not infarction, most commonly in the parieto-occipital regions. The severity of this clinical symptom varies. For example, the visual disturbance can manifest as blurred vision, homonymous hemianopsia, or even cortical blindness.Patients may be mildly confused or agitated but can become comatose.Other symptoms less commonly seen include nausea, vomiting, seizures, and brainstem deficits.Chronic kidney disease (CKD) and acute kidney injury are both commonly present in patients with PRES. We are presenting a rare case of neurogenic bladder who developed PRES after augmentation cystoplasty due to underlying CKD.
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A rare case of a common hepatic duct stricture secondary to an anteriorly crossing right hepatic artery in an infant p. 161
Aureen D’Cunha, B S. S Ravi Kishore, Isaac Tharu Varghese
A 1-year-old male child presented with progressive jaundice. Investigations were suggestive of an obstructive pathology with a suspected choledochal cyst on imaging. Intraoperative cholangiogram demonstrated runoff of contrast from the cystic duct into the common bile duct with no opacification of the biliary system proximal to this. Intraoperatively, the right hepatic artery was found anteriorly crossing the common hepatic duct (CHD) causing extrinsic compression leading to complete obstruction. Following stricture excision and anastomosis of the dilated bulbous CHD to a Roux-en-Y jejunal loop, the child recovered completely. An anteriorly crossing right hepatic artery causing obstruction to the biliary duct is a rare occurrence, more so in infancy. An excision with a hepaticojejunostomy is straight forward and curative.
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Congenital midline cervical cleft with respiratory epithelium: A rare association p. 164
Jyoti Manakchand Bothra, Harish Jayaram, Mainak Deb, Michelle D E. Padua
Congenital midline cervical cleft is a rare anomaly and is clinically apparent at birth. Histology of this defect is consistent with the presence of stratified squamous epithelium. However, we present a case of 1-year-old boy with chronic mucocutaneous candidiasis associated with two cysts and presence of focal respiratory epithelium. We attempt to discuss the presentation, the histological differences, and the optimal surgical treatment for the same.
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Penile cutaneous horn in a 22-month-old child: A rare case report p. 167
Girish Prabhakar, Akshita Bhat
Cutaneous horn is a horn-like hyperkeratotic lesion. Its presence over penis is unusual and rare. Herein, we report a case of horn of penis in a 22-month-old child.
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Amyand's hernia, appendicitis, and undescended testis: The amyand's triad p. 169
Vembar Dhanasekarapandian, Vivek Shanmugam, Muthukumaran Jagannathan
Inguinal hernia with appendix as content, classically called as Amyand's Hernia has been well documented in children. Undescended testis with an associated hernia is also not uncommon and is an indication for early orchidopexy. We report a case of undescended testis which presented as an irreducible right inguinal hernia, which on exploration was found to have an inflamed appendix.
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Needle insertion in a child: A rare form of child abuse p. 171
Archika Gupta, Om Prakash Purbey, Kanoujia Sunil, Anand Pandey, Shiv Narain Kureel
Needle insertion is a rare form of child abuse that, though prevalent in the society as an attempted infanticide/homicide, remains undiagnosed and underreported. One should have strong suspicion for occurrence of child abuse in a child with history of needle injury. Here, we report an unusual case of 3-month-old boy with history of hypodermic needle extrusion from multiple sites of body and suspicion of possible child abuse.
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Acquired ileal atresia in a 3-year-old patient: A rare case p. 174
Dileep Garg, Aditya Pratap Singh, Arun Kumar Gupta, Arvind Kumar Shukla
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Aplasia cutis congenita in a newborn: A rare case p. 175
Aditya Pratap Singh, Arun Kumar Gupta, Rajlaxmi Pardeshi, Arvind Kumar Shukla
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