Rare neonatal diabetes insipidus and associated late risks: Case report

BMC Pediatrics

Susana Serrano G-Gallarza¹  Lorena Miñones-Suárez²  Maximiliano Francisco Rivas-Crespo² 

[1] Neuroradiology, Hospital San Agustín, Avilés, Spain;Department of Pediatrics, Hospital Universitario Central de Asturias, Oviedo, Spain

关键词: Desmopressin;    Dehydration;    Myelinolysis;    Undernutrition;    Neurodevelopmental delay;    Central diabetes insipidus;    Neonatal intraventricular hemorrhage;    Coarctation of the aorta;    Infant;    At term newborn;   

Others  :  1170789
DOI  :  10.1186/1471-2431-12-56

 received in 2011-12-01, accepted in 2012-05-28,  发布年份 2012

【 摘 要 】

Background

Most cases of neonatal central diabetes insipidus are caused by an injury, which often results in other handicaps in the patient. The infant’s prognosis will be determined by his or her own early age and disability as well as by the physician’s skill. However, the rarity of this condition prevents the acquisition of personal experience dealing with it.

Case Presentation

A neonatal hemorrhagic stroke, caused by an aortic coarctation, caused right lower limb paresis, swallowing disability, and central diabetes insipidus in a term infant. The scant oral intake, as a consequence of his disability, caused progressive undernutrition which closed a vicious circle, delaying his development and his ability to overcome the swallowing handicap. On the other hand, nasal desmopressin absorption was blocked by several common colds, resulting in brain bleeding because of severe dehydration. This even greater brain damage hampered the improvement of swallowing, closing a second harmful circle. Moreover, a devastating central myelinolysis with quadriplegia, caused by an uncontrolled intravenous infusion, consummated a pernicious sequence, possibly unreported.

Conclusions

The child’s overall development advanced rapidly when his nutrition was improved by gastrostomy: This was a key effect of nutrition on his highly sensitive neurodevelopment. Besides, this case shows potential risks related to intranasal desmopressin treatment in young children.

【 授权许可】

   
2012 Rivas-Crespo et al.; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

Figure 2.

【 参考文献 】

• [1]Armstrong-Wells J, Johnston SC, Wu YW, Sidney S, Fullerton HJ: Prevalence and predictors of perinatal hemorrhagic stroke: results from the Kaiser pediatric stroke study. Pediatrics 2009, 123:823-828.
• [2]Wu YW, Hamrick SE, Miller SP, Haward MF, Lai MC, Callen PW, Barkovich AJ, Ferriero DM: Intraventricular hemorrhage in term neonates caused by sinovenous thrombosis. Ann Neurol 2003, 54:123-126.
• [3]Mehwald PS, Dittrich S, Grohmann J, Bley T, Kececioglu D: Coarctation of the aorta presenting as cerebral hemorrhage. J Pediatr 2005, 146:293.
• [4]Lee YJ, Huang FY, Shen EY, Kao HA, Ho MY, Shyur SD, Chiu NC: Neurogenic diabetes insipidus in children with hypoxic encephalopathy: six new cases and a review of the literature. Eur J Pediatr 1996, 155:245-248.
• [5]Cheetham T, Baylis PH: Diabetes Insipidus in Children. Pathophysiology, Diagnosis and Management. Pediatr Drugs 2002, 4:785-796.
• [6]Mastrangelo S, Arlota A, Cefalo MG, Maurizi P, Cianfoni A, Riccardi R: Central Pontine and Extrapontine Myelinolysis in a Pediatric Patient Following Rapid Correction of Hypernatremia. Neuropediatrics 2009, 40:144-147.
• [7]Brown WD: Osmotic demyelination disorders: central pontine and extrapontine myelinolisis. Curr Opin Neurol 2000, 13:691-697.
• [8]Venkatanarasimha N, Mukonoweshuro W, Jones J: AJR teaching file: symmetric demyelination. Am J Roentgenol 2008, 191(Suppl 3):S34-S36.
• [9]Cherian S, Whitelaw A, Thoresen M, Love S: The pathogenesis of neonatal post-hemorrhagic hydrocephalus. Brain Pathol 2004, 14:305-311.
• [10]Blanco EJ, Lane AH, Aijaz N, Blumberg D, Wilson TA: Use of Subcutaneous DDAVP in Infants with Central Diabetes Insipidus. J Ped Endocrinol Metab 2006, 19:919-925.
• [11]Ruzek KA, Campeau NG, Miller GM: Early Diagnosis of Central Pontine Myelinolysis with Diffusion-Weighted Imaging. Am J Neuroradiol 2004, 25:210-213.
• [12]De Simone T, Regna-Gladin C, Carriero MR, Farina L, Savoiardo M: Wallerian degeneration of the pontocerebellar fibers. Am J Neuroradiol 2005, 26:1062-1065.
• [13]Shah B, Tobias JD: Osmotic demyelination and hypertonic dehydration in a 9-year-old girl: changes in cerebrospinal fluid myelin basic protein. J Intensive Care Med 2006, 21:372-376.
• [14]AlOrainy IA, O’Gorman AM, Decell MK: Cerebral bleeding, infarcts, and presumed extrapontine myelinolysis in hypernatraemic dehydration. Neuroradiology 1999, 41:144-146.
• [15]Han BK, Lee M, Yoon HK: Cranial ultrasound and CT findings in infants with hypernatremic dehydration. Pediatr Radiol 1997, 27:739-742.