{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,21]],"date-time":"2025-11-21T22:09:08Z","timestamp":1763762948748,"version":"3.45.0"},"reference-count":10,"publisher":"The Pediatric Pharmacy Association","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,10]]},"abstract":"<jats:p>\n                    Congenital muscular dystrophy type 1A (MDC1A), an autosomal recessive disorder, is one of the most prevalent forms of congenital muscular dystrophy, characterized by the loss of Laminin-\u03b1\n                    <jats:sub>2<\/jats:sub>\n                    subunit (Merosin). Approximately one-third of affected patients experience epileptic seizures, typically manifesting around 8 years of age, with focal onset and secondary generalization, often with tonic-clonic semiology. Most reported cases show limited or no response to conventional treatment, though a subset responds to valproate or lamotrigine. The efficacy of levetiracetam in these patients remains insufficiently studied. Metabolic acidosis is not listed as a known adverse effect of levetiracetam in the medication\u2019s technical information. In this case, an 11-year-old male with MDC1A presented with nocturnal seizure equivalents and was started on levetiracetam therapy. Approximately 24 hours after receiving the loading dose, the patient\u2019s condition deteriorated significantly, and severe metabolic acidosis with an elevated anion gap was observed. The patient required transfer to the pediatric intensive care unit and received intensive intravenous hydration and potassium supplementation. Upon discontinuation of levetiracetam, the patient stabilized, and metabolic normalization was achieved within approximately 24 hours. There are very few reports in the literature that also point to the development of a high anion gap metabolic acidosis after levetiracetam administration. The underlying mechanism is hypothesized but not experimentally verified, and a causal relationship remains unproven. Nevertheless, this observation represents a potentially serious adverse event associated with a commonly used medication, warranting heightened clinical awareness. We therefore recommend actively highlighting this and considering safety monitoring based on the individual risk of the patients being treated.\n                  <\/jats:p>","DOI":"10.5863\/jppt-24-00127","type":"journal-article","created":{"date-parts":[[2025,11,21]],"date-time":"2025-11-21T22:04:08Z","timestamp":1763762648000},"page":"668-672","source":"Crossref","is-referenced-by-count":0,"title":["High Anion Gap Metabolic Acidosis (HAGMA) After Levetiracetam Administration in an 11-Year-Old Boy With Laminin-\u03b12-Deficiency-Associated Muscular Dystrophy and Epilepsy"],"prefix":"10.5863","volume":"30","author":[{"given":"Piet Erik","family":"Beusker","sequence":"first","affiliation":[{"name":"Department of Pediatrics, University of Bielefeld, University Hospital OWL, Children\u2019s Center Bethel, Bielefeld, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Doortje Marieke","family":"Mehrtens","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Bielefeld, University Hospital OWL, Children\u2019s Center Bethel, Bielefeld, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eckard","family":"Hamelmann","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Bielefeld, University Hospital OWL, Children\u2019s Center Bethel, Bielefeld, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Georg","family":"Classen","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Bielefeld, University Hospital OWL, Children\u2019s Center Bethel, Bielefeld, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"3883","reference":[{"key":"i2331-348X-30-5-668-b01","series-title":"Nelson Textbook of Pediatrics","volume":"Volume 2","author":"Kliegman","year":"2020","unstructured":"Kliegman RM, Nelson WE. Nelson Textbook of Pediatrics. Volume 2. 21 ed. Philadelphia, PA: Elsevier; 2020."},{"key":"i2331-348X-30-5-668-b02","article-title":"Kongenitale Myopathien","unstructured":"Gesellschaft f\u00fcr Muskelkranke. Kongenitale Myopathien. Accessed August 7, 2024. https:\/\/www.dgm.org\/muskelerkrankungen\/kongenitale-myopathien"},{"issue":"5","key":"i2331-348X-30-5-668-b03","series-title":"Epilepsia","first-page":"971","article-title":"Epilepsy in LAMA2-related muscular dystrophy: an electro-clinico-radiological characterization","volume":"61","author":"Natera-de Benito","year":"2020","unstructured":"Natera-de Benito D, Muchart J, Itzep D, Ortez C, Gonz\u00e1lez-Quereda L, Gallano P et al. Epilepsy in LAMA2-related muscular dystrophy: an electro-clinico-radiological characterization. Epilepsia. 2020;61(5):971\u2013983."},{"issue":"4","key":"i2331-348X-30-5-668-b04","series-title":"Neuromuscul Disord","first-page":"241","article-title":"Genotype-phenotype correlation in a large population of muscular dystrophy patients with LAMA2 mutations","volume":"20","author":"Geranmayeh","year":"2010","unstructured":"Geranmayeh F, Clement E, Feng LH, et al. Genotype-phenotype correlation in a large population of muscular dystrophy patients with LAMA2 mutations. Neuromuscul Disord. 2010;20(4):241\u2013250."},{"key":"i2331-348X-30-5-668-b05","series-title":"Seizure","first-page":"425","article-title":"Epilepsy in LAMA2-related muscular dystrophy: a systematic review of the literature","volume":"91","author":"Salvati","year":"2021","unstructured":"Salvati A, Bonaventura E, Sesso G, et al. Epilepsy in LAMA2-related muscular dystrophy: a systematic review of the literature. Seizure. 2021;91:425\u2013436."},{"key":"i2331-348X-30-5-668-b06","series-title":"Sun Pharmaceuticals Germany GmbH","unstructured":"Levetiracetam [package insert]. Leverkusen, Germany: Sun Pharmaceuticals Germany GmbH. Accessed August 7, 2024. https:\/\/ec.europa.eu\/health\/documents\/community-register\/2021\/20210318151210\/anx_151210_en.pdf"},{"issue":"2","key":"i2331-348X-30-5-668-b07","series-title":"Clin Pharmacol Ther","first-page":"239","article-title":"A method for estimating the probability of adverse drug reactions","volume":"30","author":"Naranjo","year":"1981","unstructured":"Naranjo CA, Busto U, Sellers EM, et al. A method for estimating the probability of adverse drug reactions. Clin Pharmacol Ther. 1981;30(2):239\u2013245."},{"key":"i2331-348X-30-5-668-b08","series-title":"LiverTox: Clinical and Research Information on Drug-Induced Liver Injury","year":"2012","unstructured":"LiverTox: Clinical and Research Information on Drug-Induced Liver Injury. Bethesda, MD: National Institute of Diabetes and Digestive and Kidney Diseases; 2012."},{"key":"i2331-348X-30-5-668-b09","article-title":"Levetiracetam and Metabolic acidosis - a phase IV clinical study of FDA data","unstructured":"eHealthMe.com. Levetiracetam and Metabolic acidosis - a phase IV clinical study of FDA data. Accessed August 7, 2024. https:\/\/www.ehealthme.com\/ds\/levetiracetam\/metabolic-acidosis\/"},{"key":"i2331-348X-30-5-668-b10","series-title":"Am J Respir Crit Care Med","first-page":"A4808","article-title":"Levetiracetam as a cause of high anion gap metabolic acidosis","volume":"199","author":"Megri","year":"2019","unstructured":"Megri MA, Abusnina WA, Bukamur HS, Shweihat YR. Levetiracetam as a cause of high anion gap metabolic acidosis. Am J Respir Crit Care Med. 2019;199:A4808."}],"container-title":["The Journal of Pediatric Pharmacology and Therapeutics"],"original-title":[],"link":[{"URL":"https:\/\/jppt.kglmeridian.com\/view\/journals\/jppt\/30\/5\/article-p668.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/jppt.kglmeridian.com\/downloadpdf\/journals\/jppt\/30\/5\/article-p668.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/jppt.kglmeridian.com\/downloadpdf\/journals\/jppt\/30\/5\/article-p668.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,11,21]],"date-time":"2025-11-21T22:04:08Z","timestamp":1763762648000},"score":1,"resource":{"primary":{"URL":"https:\/\/jppt.kglmeridian.com\/view\/journals\/jppt\/30\/5\/article-p668.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10]]},"references-count":10,"journal-issue":{"issue":"5"},"URL":"https:\/\/doi.org\/10.5863\/jppt-24-00127","relation":{},"ISSN":["2331-348X","1551-6776"],"issn-type":[{"value":"2331-348X","type":"electronic"},{"value":"1551-6776","type":"print"}],"subject":[],"published":{"date-parts":[[2025,10]]}}}