Treating hypothalamic morbid obesity with laparoscopic Roux-en-Y. gastric bypass (LRYGB) in patient with craniopharyngioma. Report

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1 1 Treating hypothalamic morbid obesity with laparoscopic Roux-en-Y gastric bypass (LRYGB) in patient with craniopharyngioma. Report of a case. Harilaos Pappis 1, Demetrios Moris 1, Dimitra Kotsakou 1, Nikolaos Pararas rd Surgical Department, Hygeia Hospital, Athens, Greece Authors H. Pappis and D. Moris equally contributed. Corresponding Author: Demetrios N. Moris. Anastasiou Gennadiou 56, 11474, Athens, Greece, dimmoris@yahoo.com, tel-fax: Running head: LRYGB in hypothalamic-related obesity Keywords: Craniopharyngioma, hypothalamic obesity, gastric bypass Article category: Case report and review of the literature.

2 2 Abstract Aim-Background: Obesity due to hypothalamic dysfunction is a severe medical situation that lacks an established treatment in the present protocol. Methods: In the present article, we describe a case of a 29 years old patient with a medical history of surgical resection of craniopharyngioma at the age of 8 years who presented in our department with hypothalamic dysfunction and hypothalamic related obesity without neurologic symptoms. After reviewing the literature till September 2012, we found 5 cases of surgical treatment of this kind of obesity with gastric bypass with similar results with our case. Results: The patient underwent a LRYGB. Postoperative course was uneventful and 2 years after the operation, the patient presents a significant weight loss, as well as diminished concomitant disorders. Conclusions: It is concluded that gastric bypass procedures offer a reliable and efficient treatment choice in cases of hypothalamic-related obesity. Keywords: Craniopharyngioma, hypothalamic obesity, gastric bypass

3 3 Introduction Obesity due to hypothalamic dysfunction is a severe medical situation that lacks an established treatment in the present protocol. Hypothalamic obesity is a potential sequela of craniopharyngioma, arising from hypothalamic damage inflicted by either the tumor and/or its treatment. The marked weight gain that characterizes this disorder appears to result from impaired sympathoadrenal activation, parasympathetic dysregulation, and other hormonal and hypothalamic disturbances that upset the balance between energy intake and expenditure. In literature, 5 cases of surgical treatment of this kind of obesity with gastric bypass are described. To our knowledge, we describe the first case in Greece and the sixth worldwide of treating hypothalamic morbid obesity with laparoscopic Roux-en-Y gastric bypass (LRYGB) in patient with craniopharyngioma. Case presentation In the present article, we describe a case of a 29 years old patient with a medical history of surgical resection of craniopharyngioma at the age of 8 years who presented in our department with hypothalamic dysfunction and hypothalamic related obesity without neurologic symptoms. From the rest medical history, the patient had a laparoscopic cholecystectomy in The patient visited our department before 2 years with a body mass index(bmi) of 38,5 (105kg, 1,65m), obesity that was developed gradually after the surgical resection of the craniopharyngioma, due to hyperphagia and increased appetite, tendency that could not be managed with repeated dietary restriction and physical exercise. Distresses that accompanied the obesity were the diabetes insipidus, sleep apnea and

4 4 joint pain. The preoperative regime included Hydrocortisone 5mg x 2, Desmopressin 120 mg x 2 and thyroid replacement therapy (0,1mg 1 ½ x 1). The patient underwent a laparoscopic Roux-en-Y gastric bypass with a 150 cm Roux limb (LRYGB). The gastrointestinal anastomosis was very demanding due to the fragility of the gastric mucosa and the lack of adequate gastric tightness. It was performed 3 times till we reached the optimal result. For the patient s safety, the conversion to an open procedure was decided, and the anastomosis was completed in hand (it was a unique case of conversion in our series of 52 laparoscopic gastric bypasses). Follow-up The postoperative follow up was scheduled every 3 months in the first year and every 6 months during the second one, including extensive biochemical testing, vitamin profile as well as gastroscopy every 6 months. No pathological value or observation occurred. 2 years after the operation, the patient presents a significant weight loss (75kg, ΒΜΙ 27,5), as well as diminished concomitant disorders. Sleep apnea had declined and the urge to eat had decreased significantly. By replacing the vitamin deficit with a multivitamin concoction during the first postoperative year, dietary disorders or insufficiencies were prevented. After the loss of weight, the dosage of Hydrocortisone remained in the same, low levels, without signs of adrenal insufficiency. Discussion Patients with childhood-onset craniopharyngioma often suffer from obesity[1]. This obesity syndrome is characterized by a rapid weight gain that may be accompanied

5 5 by severe hyperphagia. Weight gain occurs from the disruption of the normal homeostatic functioning of the hypothalamic centers responsible for controlling satiety and hunger and regulating energy balance that results to hyperphagia, autonomic imbalance, reduction of energy consuming and hyperinsulinemia[2].thus weight increase has traditionally been refractory to usual dietary and lifestyle interventions. Two important etiological factors of obesity development are mostly discussed in literature: energy intake and physical activity. Energy intake was supposed to be high due to a disturbed hypothalamic regulation of appetite. But it was suggested that reduced physical activity, rather than increased energy intake, in patients with craniopharyngioma is responsible for the obesity development noted in these subjects [3]. Inge et al [4] have first published a case of 14 years old extremely obese boy (BMI>60 kg/m 2 ) treated with gastric bypass surgery. Its obesity was attributed to hypothalamic dysfunction after the resection of a childhood-onset craniopharyngioma. In this patient, dietary interventions were unsuccessful, and pharmacologic intervention only slowed the rate of weight gain, clinical outcome that is similar to our case. On the other hand, gastric bypass surgery led to reduced food urges, significant weight loss, amelioration of obesity-related comorbidities as well as homeostasis to the hormone status including normalization of fasting hyperinsulinemia, postprandial insulin responses and reductions in active ghrelin and leptin concentrations[3], results that are in the same tone with ours. Rottembourg et al [5] have recently published two cases of adolescents treated with bariatric surgery procedures for their hypothalamic obesity, due to childhood onset craniopharyngioma, non responding to sustained nutritional and exercise-oriented interventions. It was concluded that in these patients, bariatric surgery seems to be an efficacious medical option available for hypothalamic obesity [5]. Schultes et al [2] presented a case of a

6 6 29-year-old obese man, who had undergone craniopharyngioma resection at the age of 8, with a BMI of 52.0 kg/m 2, type 2 diabetes and obstructive sleep apnea syndrome. The patient underwent a distal gastric bypass operation and presented a significant compliance with his treatment. The results after 18 months of follow up, were similar to ours and include: BMI had decreased to 31.9 kg/m 2, type 2 diabetes was in complete remission and sleep apnea appeared to be improved [2]. Recently Page- Wilson et al [6] published a case of 18-year-old Caucasian female with a history of craniopharyngioma resection who presented with rapid weight gain. Her weight was 101 kg, height 62 inches (mid-parental height 66 inches) and BMI 40.8 kg/m 2. The patient underwent an uncomplicated Roux en Y gastric by-pass with a 150 cm Roux limb. Nine months after bypass her weight had decreased to 93.9 kg (BMI 39.0) and remained relatively stable 19 months post-bypass. Fasting leptin and insulin levels decreased from baseline and the circulating ghrelin levels increased from baseline [6]. Table 1 summarizes the review of the literature on the subject. Conclusions After being treated for the craniopharyngioma, these patients present a tendency to become obese [1] or even to develop metabolic syndrome [7]. As weight gain starts early after diagnosis and severe obesity causes a reduction in quality of life, early therapeutic efforts should be considered in these patients. In cases of hypothalamic related morbid obesity without neurologic symptoms, that is difficult to respond to physical exercise or diet or even pharmacologic intervention, long loop gastric bypass presents as an exceptional approach and therapeutic choice with the contribution and collaboration of physicians of different specialties and expertise. At present, the

7 7 published cases of bariatric surgery for the treatment of hypothalamic obesity secondary to craniopharyngioma suggest that surgical weight loss interventions can be safely and effectively used for the management of this disease, as it appears to not only decrease body mass but to attenuate appetite even in the presence of hypothalamic damage. Importantly, these benefits are also accompanied by improvements in reported quality of life. Conflict of Interest No conflicts of interest declared. References 1. Iughetti L, Bruzzi P. Obesity and craniopharyngioma. Ital J Pediatr 2011;37: Schultes B, Ernst B, Schmid F, Thurnheer M. Distal gastric bypass surgery for the treatment of hypothalamic obesity after childhood craniopharyngioma. Eur J Endocrinol 2009;161: Harz KJ, Muller HL, Waldeck E, Pudel V, Roth C. Obesity in patients with craniopharyngioma: assessment of food intake and movement counts indicating physical activity. J Clin Endocrinol Metab 2003;88: Inge TH, Pfluger P, Zeller M, Rose SR, Burget L, Sundararajan S, et al. Gastric bypass surgery for treatment of hypothalamic obesity after craniopharyngioma therapy. Nat Clin Pract Endocrinol Metab 2007;3:

8 8 5. Rottembourg D, O'Gorman CS, Urbach S, Garneau PY, Langer JC, Van Vliet G, et al. Outcome after bariatric surgery in two adolescents with hypothalamic obesity following treatment of craniopharyngioma. J Pediatr Endocrinol Metab 2009;22: Page-Wilson G, Wardlaw SL, Khandji AG, Korner J. Hypothalamic obesity in patients with craniopharyngioma: treatment approaches and the emerging role of gastric bypass surgery. Pituitary 2012;15: Sahakitrungruang T, Klomchan T, Supornsilchai V, Wacharasindhu S. Obesity, metabolic syndrome, and insulin dynamics in children after craniopharyngioma surgery. Eur J Pediatr 2010;170: Table 1. A summary of the cases of hypothalamic-related obesity treated with bariatric surgery procedures. Author Year Number Procedure Results of cases Inge et al [4] Gastric bypass Gastric bypass surgery led to reduced food urges, significant weight loss, amelioration of obesity-related comorbidities as well as homeostasis to the hormone status Rottembourg et al [5] Bariatric surgery(specific procedure not available) It was concluded that in these patients, bariatric surgery seems to be an efficacious medical option available for hypothalamic

9 9 obesity Schultes et al [2] Distal gastric bypass After 18 months of follow up, body mass index (BMI) had decreased to 31.9 kg/m(2), type 2 diabetes was in complete remission and sleep apnea appeared to be improved. Page-Wilson et al [6] Roux en Y gastric bypass with a 150 cm Roux limb(lrygb) 9 months after bypass her weight had decreased to 93.9 kg (BMI 39.0) and remained relatively stable 19 months post-bypass. Fasting leptin and insulin levels decreased from baseline and the circulating ghrelin levels increased from baseline.

10 10 Περίληψη Σκοπός: Η παχυσαρκία που προκύπτει από βλάβη στον υποθάλαμο, είναι μια σοβαρή κατάσταση που στερείται επί του παρόντος πρωτοκόλλου τεκμηριωμένης θεραπείας. Στην παγκόσμια βιβλιογραφία αναφέρονται 5 περιστατικά χειρουργικής αντιμετώπισης ασθενών με υποθαλαμικού τύπου νοσογόνο παχυσαρκία με γαστρική παράκαμψη. Μέθοδοι: Στο παρόν άρθρο αναφέρουμε την εμπειρία μας με μια 29χρονη ασθενή με χειρουργηθέν κρανιοφαρυγγίωμα σε ηλικία 8 ετών με υποφυσιακή ανεπάρκεια, χωρίς νευρολογική συνδρομή και υποθαλαμικού τύπου παχυσαρκία. Μετά από θεραπεία για το κρανιοφαρυγγίωμα, οι ασθενείς αυτοί παρουσιάζουν τάση για παχυσαρκία. Αποτελέσματα: Η ασθενής υπεβλήθη σε λαπαροσκοπική επέμβαση γαστρικής παράκαμψης μακριάς έλικας. Η γαστρο-εντεροαναστόμωση χρειάστηκε να επαναληφθεί 3 φορές λόγω ευθριπτότητας του βλεννογόνου του στομάχου και κακής στεγανότητας. Για την ασφάλεια της ασθενούς αποφασίστηκε μετατροπή της επέμβασης σε ανοιχτή και η αναστόμωση τελείωσε στο χέρι. (Μοναδική περίπτωση μετατροπής στη σειρά μας που αριθμεί 52 λαπαροσκοπικές γαστρικές παρακάμψεις). Η μετεγχειρητική παρακολούθηση έγινε κάθε 3 μήνες τον 1 ο χρόνο και κάθε 6 μήνες το 2 ο με πλήρη βιοχημικό έλεγχο και πλήρες προφίλ βιταμινών καθώς και γαστροσκόπηση κάθε 6 μήνες. Δεν παρατηρήθηκε καμία παθολογία στις παραπάνω εξετάσεις. 2 χρόνια μετά το χειρουργείο παρουσιάζει σημαντική μείωση του σωματικού βάρους (75kg, ΒΜΙ 27,5), καθώς και των συνοδών της προβλημάτων. Η υπνική άπνοια έχει εξαληφθεί και η αίσθηση της πείνας έχει σημαντικά μειωθεί.

11 11 Συμπεράσματα: Μετά από θεραπεία για το κρανιοφαρυγγίωμα, οι ασθενείς αυτοί παρουσιάζουν τάση για παχυσαρκία. Σε περιπτώσεις ασθενών με υποθαλαμικού τύπου νοσογόνο παχυσαρκία και χωρίς νευρολογική συνδρομή, οι οποίοι είναι πολύ δύσκολο να ανταποκριθούν σε απώλεια βάρους με δίαιτα, γυμναστική ή ακόμα και φαρμακευτική αγωγή, με τη συνεργασία ιατρών πολλών ειδικοτήτων, η επέμβαση γαστρικής παράκαμψης μακριάς έλικας μπορεί να αποτελέσει μια εξαιρετική λύση.

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