Central pontine myelinolysis and locked‐IN syndrome associated with tacrolimus after pediatric heart transplantation

Background Locked‐in syndrome represents the most severe form of central pontine myelinolysis and varies in presentation from asymptomatic to fully developed locked‐in‐syndrome characterized by the combination of quadriplegia, loss of the ability to communicate except through the use of the eyes, an...

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Bibliographic Details
Published in:Pediatric transplantation 2023-05, Vol.27 (3), p.e14474-n/a
Main Authors: Albert‐Brotons, Dimpna C., Alharkan, Waleed, Beheri, Reem, AlHaddad, Mohammed, Alali, Yasser, Alheraish, Yasser, Chedrawi, Aziza, AlHalees, Zohair
Format: Article
Language:English
Subjects:
Calcineurin inhibitors
central pontine myelinolysis
Child
Demyelination
heart transplant
Heart transplantation
Heart Transplantation - adverse effects
Humans
Immunosuppression
Locked-In Syndrome
Magnetic Resonance Imaging
Male
Myelin
Myelinolysis, Central Pontine
Neurological complications
Pediatrics
Rapamycin
Sirolimus
Spontaneous recovery
Tacrolimus
Tacrolimus - adverse effects
TOR protein
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Summary:Background Locked‐in syndrome represents the most severe form of central pontine myelinolysis and varies in presentation from asymptomatic to fully developed locked‐in‐syndrome characterized by the combination of quadriplegia, loss of the ability to communicate except through the use of the eyes, and an inability to follow commands. Methods We report a 10‐year‐old boy who developed a severe case of locked‐in syndrome after heart transplantation. Results Patient had a spontaneous recovery, treated with supportive treatment and the improvement was detected with cessation of calcineurin inhibitor therapy by substituting with an mTOR inhibitor (sirolimus). No cases of locked‐in syndrome post‐heart transplant in pediatrics cases have been documented in the literature. Conclusion Physicians should recognize a rapid progression of central pontine myelinolysis and locked‐in syndrome in the context of heart transplant and although several factors likely contributed to this outcome, adjustment of immunosuppression including by substituting tacrolimus with sirolimus could be effective.
ISSN:1397-3142
1399-3046
DOI:10.1111/petr.14474