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Year : 2020  |  Volume : 15  |  Issue : 3  |  Page : 308-310

Recurrent urinary tract infection in craniopharyngioma: A harbinger to a sinister diagnosis!

Department of Pediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India

Date of Submission06-Feb-2020
Date of Decision30-May-2020
Date of Acceptance03-Jul-2020
Date of Web Publication06-Nov-2020

Correspondence Address:
Dr. Aashima Dabas
Department of Pediatrics, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi.
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jpn.JPN_20_20

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Craniopharyngiomas are benign tumors of neuroepithelial origin, believed to arise from remnants of Rathke’s pouch. Their proximity to vital structures of the visual pathway and hypothalamus leads to both neurological and endocrinological complications. Endocrinal complications are seen in 40%–87% of the affected and can develop at presentation or post-surgery and radiotherapy. Central diabetes insipidus (CDI) is a common endocrinopathy associated with craniopharyngioma, but rarely a presenting symptom before or after surgery. CDI most commonly presents with polyuria and polydipsia. Here, we report a postoperative child with craniopharyngioma where recurrent urinary tract infection and hydronephrosis were initial clues to diagnose CDI.

Keywords: Central diabetes insipidus, endocrine disorder, pituitary hormone deficiency, polyuria

How to cite this article:
Dabas A, Yadav S, Jha M. Recurrent urinary tract infection in craniopharyngioma: A harbinger to a sinister diagnosis!. J Pediatr Neurosci 2020;15:308-10

How to cite this URL:
Dabas A, Yadav S, Jha M. Recurrent urinary tract infection in craniopharyngioma: A harbinger to a sinister diagnosis!. J Pediatr Neurosci [serial online] 2020 [cited 2022 Nov 29];15:308-10. Available from: https://www.pediatricneurosciences.com/text.asp?2020/15/3/308/300052

   Introduction Top

Multiple pituitary hormone deficiency is a complication of craniopharyngioma that can be seen both due to local invasion at presentation and/or as a complication of therapy.[1] The probability of occurrence of any endocrinal disturbance is higher in children than adults,[2] and is higher in the postoperative period (64%–100% cases) than at initial diagnosis (40%–87%) in children.[3] Most case series do not document normalization of preoperative endocrine deficiency after treatment in craniopharyngioma, irrespective of the treatment modality used.[1],[4] Growth failure, hypogonadism, hypothyroidism, and hypocortisolism are commonly reported in survivors of craniopharyngioma.[1],[4],[5] Central diabetes insipidus (CDI) is seen in 65%–96% childhood craniopharyngioma survivors,[3] such that permanent CDI is more likely than transient CDI in pediatric than adult patients.[2] A high index of suspicion is required to detect CDI early. We present a case of delayed diagnosis of CDI where recurrent urinary tract infections (UTIs) helped in detection.

   Case Report Top

An 18-year-old boy presented to pediatric outpatient department with increased urinary frequency and urge incontinence for last 10 days. He complained of recurrent similar episodes for the last 2 years, which resolved on oral antibiotics. There was no history of fever, hematuria, or abdominal pain. Urine examination showed the presence of pus cells with positive culture for  Escherichia More Details coli, suggestive of UTI. An ultrasound of the kidneys revealed bilateral mild hydronephrosis (right kidney 8.3 × 3.2 cm, left kidney 9 × 4.8 cm) with bladder wall thickened 8.8mm and post-void residue 42 cc. The patient received oral ciprofloxacin as per culture sensitivity to which he responded. Review of records revealed that the child was diagnosed with craniopharyngioma at 10 years of age when he presented with headache and visual loss. He underwent frontotemporal craniectomy with orbitozygomatic osteotomy and tumor decompression. There were no endocrinal disorders in the preoperative period. A repeat left retromastoid craniotomy with tumor debulking was done after 1 year due to tumor recurrence, followed by 35 cycles of radiotherapy. The patient remained free of any neurological or systemic complaints till 16 years of age when he developed recurrent UTI and received multiple courses of oral antibiotics elsewhere. History on review confirmed the presence of polyuria and nocturnal enuresis in between the UTI episodes, which were the initial clues to CDI but were missed. The patient also reported increased lethargy and somnolence during the daytime, and hence had stopped attending school for the past 3 years.

At 18 years of age, his weight was 56kg (−0.49 standard deviation score (SDS)), height was 160.5 cm (−1.69 SDS), body mass index (BMI) was 21.74kg/m2 (+0.22 SDS), and bilateral testicular volume was 2mL with absent pubic hair, and he was normotensive. The laboratory investigations showed hemoglobin at 11.6g/dL, blood urea at 21 mg/dL, serum creatinine at 0.9 mg/dL, sodium at 146 meq/L, and potassium at 4.2 meq/L, and normal liver function tests with a skeletal age of 14 years (Tanner Whitehouse III method). Further investigations revealed hypocortisolism (serum cortisol, 17.2 nmol/L), central hypothyroidism (thyroid-stimulating hormone [TSH], 7.5 mIU/L, free T4, 10.2 pmol/L, and free T3, 4.8 pmol/L), and hypogonadism (serum luteinizing hormone [LH], <0.1IU/L, follicle-stimulating hormone [FSH], 1.3IU/L, and testosterone, 1.2ng/dL), suggestive of multiple pituitary hormonal deficiencies. CDI was confirmed on water deprivation test (serum osmolality, 300 mOsm/kg, urine osmolality, 133 mOsm/kg; 479 mOsm/kg after desmopressin). A repeat magnetic resonance imaging (MRI) of the brain showed absence of tumor with an empty sella and absent pituitary bright spot. The patient was started on hydrocortisone (at 6.5 mg/m2/day) followed by thyroxine 50 ug/day, tablet desmopressin 0.1 mg/day, and monthly testosterone depot injections.

He reported improvement in urinary symptoms and malaise within 1 month of treatment. However, after 4 months, signs of hypothalamic obesity (weight, 60kg, height, 160.8 cm, and BMI, +61 SDS) began to evolve with symptoms of polyphagia for which dietary measures were instituted. After 12 months, hydronephrosis had decreased (right kidney, 8 × 3.1 cm, left kidney, 9 × 3.4 cm, bladder wall thickness, 5mm, and post-void residue, 10 cc) with no repeat episode of UTI or enuresis. His weight, however, increased to 70kg, height to 162.5 cm, and BMI to 26.51kg/m2 (+1.35 SDS) with early fatty changes in the liver, suggestive of metabolic syndrome. Poor height velocity confirmed coexistent growth hormone (GH) deficiency (peak GH, 1.2ng/dL on provocative testing). The patient reported 2kg weight loss after strict dietary measures in the last follow-up visit after 18 months, and he continues on medical therapy.

   Discussion Top

The common causes of CDI in childhood are brain malformations and nonstructural intracranial pathologies such as meningitis, tumor, and histiocytosis.[6],[7],[8] Idiopathic CDI is a rare diagnosis where an underlying pathology may be discernible after first few years of diagnosis.[9] Most patients with CDI have associated anterior pituitary defect detectable at presentation[6] or may evolve later.[7] Presence of pituitary stalk thickness or absent pituitary on neuroimaging predicts concurrent anterior pituitary defect as was also seen in the index case.[9]

CDI in craniopharyngioma is a common occurrence, which manifests as polyuria and polydipsia. However, majority of pediatric subjects with CDI report anterior pituitary defects other than polyuria and polydipsia at presentation.[6],[10] A review of 36 cases of craniopharyngioma concluded that both growth deceleration and CDI are frequently overlooked complaints, and CDI was more prevalent than actually reported.[5] Hypothalamic obesity is frequently reported complication in postoperative period when associated with radiotherapy as seen in the index case.[2]

The presentation of CDI with hydronephrosis is rarely reported.[11],[12] The polyuria associated with CDI may increase bladder pressure and cause functional obstruction of the draining system.[13] The overdistended bladder is associated with dilatation of urinary tracts and urinary stasis, which can predispose to recurrent UTI as seen in the index case.

The occurrence of endocrinopathy in craniopharyngioma increases with increase in median duration of follow-up in postoperative period.[4] The median delay in diagnosing pediatric CDI varies from 6 to 22 months,[6],[10] with median age at diagnosis between 4.7 and 10 years.[6],[7] The index patient had a significant delay in the diagnosis of CDI, which manifested after 6 years of surgery, emphasizing the need for developing a long-term follow-up and monitoring plan with the family before discharge.

To conclude, endocrinopathies are usually associated with craniopharyngioma, which should be suspected and investigated both, before and after surgery. A high index of suspicion is required to identify pediatric CDI. Common renal symptoms may be the harbinger of a serious underlying disease in pediatric craniopharyngioma survivors, and should not be ignored.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Chakrabarti I, Amar AP, Couldwell W, Weiss MH Long-term neurological, visual, and endocrine outcomes following transnasal resection of craniopharyngioma. J Neurosurg 2005;102:650-7.  Back to cited text no. 1
Capatina C, Vintila M, Gherlan I, Dumitraşcu A, Caragheolgheopol A, Procopiuc C, et al. Craniopharyngioma- clinical and therapeutic data in a mixed cohort of adult and pediatric cases. Acta Endocrinologica 2018;14:549-55.  Back to cited text no. 2
Jensterle M, Jazbinsek S, Bosnjak R, Popovic M, Zaletel LZ, Vesnaver TV, et al. Advances in the management of craniopharyngioma in children and adults. Radiol Oncol 2019;53:388-96.  Back to cited text no. 3
Karavitaki N, Brufani C, Warner JT, Adams CBT, Richards P, Ansorge O, et al. Craniopharyngiomas in children and adults: systematic analysis of 121 cases with long-term follow up. Clin Endocrinol 2005;62:397-409.  Back to cited text no. 4
de Vries L, Lazar L, Phillip M Craniopharyngioma: presentation and endocrine sequelae in 36 children. J Pediatr Endocrinol Metab 2003;16:703-10.  Back to cited text no. 5
Liu SY, Tung YC, Lee CT, Liu HM, Peng SF, Wu MZ, et al. Clinical characteristics of central diabetes insipidus in Taiwanese children. J Formos Med Assoc 2013;112:616-20.  Back to cited text no. 6
Hunter JD, Calikoglu AS Etiological and clinical characteristics of central diabetes insipidus in children: a single center experience. Int J Pediatr Endocrinol 2016;2016:3.  Back to cited text no. 7
Werny D, Elfers C, Perez FA, Pihoker C, Roth CL Pediatric central diabetes insipidus: brain malformations are common and few patients have idiopathic disease. J Clin Endocrinol Metab 2015;100:3074-80.  Back to cited text no. 8
Di Iorgi N, Allegri AE, Napoli F, Calcagno A, Calandra E, Fratangeli N, et al. Central diabetes insipidus in children and young adults: etiological diagnosis and long-term outcome of idiopathic cases. J Clin Endocrinol Metab 2014;99:1264-72.  Back to cited text no. 9
Liu W, Wang L, Liu M, Li G Pituitary morphology and function in 43 children with central diabetes insipidus. Int J Endocrinol 2016;2016:6365830.  Back to cited text no. 10
Ichiyanagi N, Gotou S, Suzuki S [Nonobstructive urinary tract dilatation due to diabetes insipidus in a patient with craniopharyngioma]. Hinyokika Kiyo 1995;41:609-12.  Back to cited text no. 11
Kruse K [Regression of a massive hydronephrosis in familial central diabetes insipidus treated with adiuretin]. Monatsschr Kinderheilkd 1984;132:612-4.  Back to cited text no. 12
van Lieburg AF, Knoers NV, Monnens LA Clinical presentation and follow-up of 30 patients with congenital nephrogenic diabetes insipidus. J Am Soc Nephrol 1999;10:1958-64.  Back to cited text no. 13

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