Scientific Letter
Diabetic ketoacidosis in an uncommon setting: management and neuromonitoring in a neonate
Cetoacidosis diabética en un escenario infrecuente. Manejo y neuromonitorización en un neonato
Beatriz González Gómeza,
, Beatriz Corredor Andrésb, Marta González-Valcárcel Espinosaa, Arturo Hernández de Bonisa, Cristina Herráiz Pereaa, Ángel Campos-Barrosc
Corresponding author
a Servicio de Neonatología, Pediatría y sus Áreas Específicas, Hospital Universitario de Toledo, Toledo, Spain
b Servicio de Endocrinología Pediátrica, Pediatría y sus Áreas Específicas, Hospital Universitario de Toledo, Toledo, Spain
c Instituto de Genética Médica y Molecular (INGEMM), IdiPAZ, Hospital Universitario La Paz, Centro deInvestigación Biomédica en Red de Enfermedades Raras (CIBERER, U-753), Instituto de Salud Carlos III, Madrid, Spain
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Manejo y neuromonitorización en un neonato" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Neonatal diabetes mellitus (NDM) manifests with persistent hyperglycaemia in the first 6 months of life. It is an infrequent diagnosis (incidence, 1:90 000–1:160 000 live births<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a>). The presentation at onset varies, ranging from asymptomatic to diabetic ketoacidosis (DKA).<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> The main complication of DKA is cerebral oedema, although poorly controlled hyperglycaemia has also been associated with neurologic and cognitive deficits.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a male neonate with adequate weight (3815 g; z score, 0.92), with no history of interest, admitted at 24 h post birth due to respiratory distress, pallor and lethargy. The vital signs were normal. Respiratory support was initiated with nasal continuous positive airway pressure, in addition to antibiotherapy. The patient required volume expansion with isotonic saline solution. The initial laboratory tests evinced hyperglycaemia (>685 mg/dL), metabolic acidosis (pH, 7.10; bicarbonate, 3.8 mmol/L) and ketonaemia (serum ketones, 6.1 mmol/L). The initial workup included blood tests, microbiological tests, lumbar puncture and toxicology testing.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Due to the suspicion of DKA, having ruled out other possible causes of hyperglycaemia, we initiated intravenous insulin therapy at a dose of 0.025 IU/kg/hour. The suspicion was confirmed by the following results: beta-hydroxybutyrate level, 8 nmol/L (<1 nmol/L); plasma insulin below 0.2 µIU/mL (2.6–24.9 µIU/mL); C-peptide, 0.10 ng/mL (1.10–4.40 ng/mL); free fatty acids, 1.88 mmol/L (0.10−0.60 mmol/L).</p><p id="par0020" class="elsevierStylePara elsevierViewall">In relation to fluid therapy, the patient developed osmotic diuresis, yet did not have clinically significant dehydration. He lost 6.5% of his birth weight, which was within the range of physiological weight loss in the first hours of life. Therefore, we calculated the fluid replacement rate to compensate for the diuresis past 2 mL/kg/h. After 6 h of infusion of isotonic saline solution, glucose was added to maintain blood glucose levels in the normal range (150−200 mg/dL). The patient did not develop any electrolyte imbalances, and required 1.5 mEq/kg/day of potassium acetate.</p><p id="par0025" class="elsevierStylePara elsevierViewall">We remained watchful for the development of clinical signs of cerebral oedema. We monitored the regional cerebral oxygen saturation by means of near-infrared spectroscopy (NIRS) and repeated head ultrasounds. The cerebral oxygen saturation values obtained by NIRS were of approximately 80%, without significant changes during the care episode.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The transcranial doppler ultrasound scan evinced increased peak systolic and end-diastolic velocities of 48.3 and 20.8 cm/s, respectively (normal values, ∼39.5 cm/s and ∼11.4 cm/s) with a decreased resistance index of 0.57 (0.6−0.8).</p><p id="par0035" class="elsevierStylePara elsevierViewall">The ketoacidosis resolved after 20 h of intravenous insulin therapy (maximum, 0.08 IU/kg/h). At 12 days, the patient started treatment with continuous subcutaneous insulin infusion. The illness had resolved completely by age 6 months, and turned out to be a transient form of NDM. He did not develop any complications and his neurodevelopment was normal.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The diagnostic evaluation included an echocardiogram and an abdominal ultrasound examination, both of which were normal. Genetic testing did not detect pathogenic variants associated with NDM.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Neonatal diabetes is infrequent, and its diagnosis requires a high level of suspicion.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> In our patient, the presence of hyperglycaemia, ketonaemia and metabolic acidosis suggested the onset of diabetes, which responded well to insulin therapy.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The main complication of DKA is cerebral oedema. It is clinically significant in 0.3%–1% of patients, in which case it is associated with a high mortality (20%–30%), and it is more likely the more severe the DKA.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Initial hypotheses on its aetiology proposed that cerebral oedema was secondary to the abrupt change in serum osmolality on treatment initiation; later, the Pediatric Emergency Care Applied Research Network (PECARN) group demonstrated that the fluid administration rate was not associated with the rate of brain injury.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Furthermore, there is evidence that in many cases, brain injury is already present at the time of treatment initiation.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Therefore, cerebral oedema would not be the cause but the consequence of brain injury.</p><p id="par0060" class="elsevierStylePara elsevierViewall">New hypotheses regarding cerebral oedema in the context of DKA suggest that oedema would develop through a complex pathophysiological mechanism involving cytotoxic, ischaemia-reperfusion and inflammatory processes, resulting in impaired cerebrovascular autoregulation and a decreased brain oxygen consumption.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">This would be consistent with the high values obtained by NIRS. Glaser et al. used NIRS to monitor patients with DKA. Eighty-seven percent of patients had regional cerebral oxygen saturation values greater than 80%, independent of the fluid administration rate. In addition, these high values persisted for hours after DKA had been resolved.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> This is also what we observed in our patient.</p><p id="par0070" class="elsevierStylePara elsevierViewall">In our patient, another Doppler ultrasound finding was consistent with the impairment of cerebrovascular autoregulation. The increases in the peak systolic and end-diastolic velocities, in addition to the decreased resistance, were evidence of cerebral hyperaemia. This supports the hypothesis of ischaemia-reperfusion being involved in the development of brain injury, as the features were the same as those found in infants with cerebral reperfusion injury.</p><p id="par0075" class="elsevierStylePara elsevierViewall">In conclusion, we present the case of a patient with a clinical presentation that is rare in the neonatal period, with evidence of impaired cerebrovascular autoregulation during the follow-up, in spite of which the patient did not develop clinically relevant brain injury or any associated sequelae.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">This research did not receive any external funding.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflicts of interest" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Neonatal diabetes mellitus: an update on diagnosis and management" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M.B. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 39 | 35 | 74 |
| 2024 September | 63 | 28 | 91 |
| 2024 August | 82 | 70 | 152 |
| 2024 July | 57 | 27 | 84 |
| 2024 June | 75 | 24 | 99 |
| 2024 May | 46 | 50 | 96 |
| 2024 April | 39 | 35 | 74 |
| 2024 March | 50 | 17 | 67 |
| 2024 February | 45 | 24 | 69 |
| 2024 January | 28 | 32 | 60 |
| 2023 December | 29 | 28 | 57 |
| 2023 November | 46 | 49 | 95 |
| 2023 October | 41 | 33 | 74 |
| 2023 September | 39 | 37 | 76 |
| 2023 August | 50 | 24 | 74 |
| 2023 July | 105 | 41 | 146 |
| 2023 June | 69 | 65 | 134 |
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