Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study

Konrad M. Szymanski; Cyrus M. Adams; Mohammad Y. Alkawaldeh; Paul F. Austin; Robin M. Bowman; Heidi Castillo; Jonathan Castillo; David I. Chu; Carlos R. Estrada; Michele Fascelli; Dominic C. Frimberger; Patricio C. Gargollo; Dawud G. Hamdan; Sarah L. Hecht; Betsy Hopson; Douglas A. Husmann; Micah A. Jacobs; Andrew E. MacNeily; Daryl J. McLeod; Peter D. Metcalfe; Theresa Meyer; Rosalia Misseri; Joseph O'Neil; Adam J. Rensing; Jonathan C. Routh; Kyle O. Rove; Kathleen J. Sawin; Bruce J. Schlomer; Isaac Shamblin; Rebecca L. Sherlock; Gennady Slobodov; Jennifer Stout; Stacy T. Tanaka; Dana A. Weiss; John S. Wiener; Hadley M. Wood; Elizabeth B. Yerkes; Jeffrey Blount

doi:10.3233/PRM-220086

Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study

Konrad M. Szymanski, Cyrus M. Adams, Mohammad Y. Alkawaldeh, Paul F. Austin, Robin M. Bowman, Heidi Castillo, Jonathan Castillo, David I. Chu, Carlos R. Estrada, Michele Fascelli, Dominic C. Frimberger, Patricio C. Gargollo, Dawud G. Hamdan, Sarah L. Hecht, Betsy Hopson, Douglas A. Husmann, Micah A. Jacobs, Andrew E. MacNeily, Daryl J. McLeod, Peter D. MetcalfeTheresa Meyer, Rosalia Misseri, Joseph O'Neil, Adam J. Rensing, Jonathan C. Routh, Kyle O. Rove, Kathleen J. Sawin, Bruce J. Schlomer, Isaac Shamblin, Rebecca L. Sherlock, Gennady Slobodov, Jennifer Stout, Stacy T. Tanaka, Dana A. Weiss, John S. Wiener, Hadley M. Wood, Elizabeth B. Yerkes, Jeffrey Blount

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Abstract

PURPOSE: This study aimed to analyze organ system-based causes and non-organ system-based mechanisms of death (COD, MOD) in people with myelomeningocele (MMC), comparing urological to other COD. METHODS: A retrospective review was performed of 16 institutions in Canada/United States of non-random convenience sample of people with MMC (born > = 1972) using non-parametric statistics. RESULTS: Of 293 deaths (89% shunted hydrocephalus), 12% occurred in infancy, 35% in childhood, and 53% in adulthood (documented COD: 74%). For 261 shunted individuals, leading COD were neurological (21%) and pulmonary (17%), and leading MOD were infections (34%, including shunt infections: 4%) and non-infectious shunt malfunctions (14%). For 32 unshunted individuals, leading COD were pulmonary (34%) and cardiovascular (13%), and leading MOD were infections (38%) and non-infectious pulmonary (16%). COD and MOD varied by shunt status and age (p < = 0.04), not ambulation or birthyear (p > = 0.16). Urology-related deaths (urosepsis, renal failure, hematuria, bladder perforation/cancer: 10%) were more likely in females (p = 0.01), independent of age, shunt, or ambulatory status (p > = 0.40). COD/MOD were independent of bladder augmentation (p = >0.11). Unexplained deaths while asleep (4%) were independent of age, shunt status, and epilepsy (p >= 0.47). CONCLUSION: COD varied by shunt status. Leading MOD were infectious. Urology-related deaths (10%) were independent of shunt status; 26% of COD were unknown. Life-long multidisciplinary care and accurate mortality documentation are needed.

Original language	English (US)
Pages (from-to)	605-619
Number of pages	15
Journal	Journal of Pediatric Rehabilitation Medicine
Volume	16
Issue number	4
DOIs	https://doi.org/10.3233/PRM-220086
State	Published - Dec 26 2023

Keywords

Myelomeningocele
cause of death
hydrocephalus
mortality
spinal dysraphism

ASJC Scopus subject areas

General Medicine

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10.3233/PRM-220086

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Szymanski, K. M., Adams, C. M., Alkawaldeh, M. Y., Austin, P. F., Bowman, R. M., Castillo, H., Castillo, J., Chu, D. I., Estrada, C. R., Fascelli, M., Frimberger, D. C., Gargollo, P. C., Hamdan, D. G., Hecht, S. L., Hopson, B., Husmann, D. A., Jacobs, M. A., MacNeily, A. E., McLeod, D. J., ... Blount, J. (2023). Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study. Journal of Pediatric Rehabilitation Medicine, 16(4), 605-619. https://doi.org/10.3233/PRM-220086

Szymanski, KM, Adams, CM, Alkawaldeh, MY, Austin, PF, Bowman, RM, Castillo, H, Castillo, J, Chu, DI, Estrada, CR, Fascelli, M, Frimberger, DC, Gargollo, PC, Hamdan, DG, Hecht, SL, Hopson, B, Husmann, DA, Jacobs, MA, MacNeily, AE, McLeod, DJ, Metcalfe, PD, Meyer, T, Misseri, R, O'Neil, J, Rensing, AJ, Routh, JC, Rove, KO, Sawin, KJ, Schlomer, BJ, Shamblin, I, Sherlock, RL, Slobodov, G, Stout, J, Tanaka, ST, Weiss, DA, Wiener, JS, Wood, HM, Yerkes, EB & Blount, J 2023, 'Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study', Journal of Pediatric Rehabilitation Medicine, vol. 16, no. 4, pp. 605-619. https://doi.org/10.3233/PRM-220086

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title = "Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study",

abstract = "PURPOSE: This study aimed to analyze organ system-based causes and non-organ system-based mechanisms of death (COD, MOD) in people with myelomeningocele (MMC), comparing urological to other COD. METHODS: A retrospective review was performed of 16 institutions in Canada/United States of non-random convenience sample of people with MMC (born > = 1972) using non-parametric statistics. RESULTS: Of 293 deaths (89% shunted hydrocephalus), 12% occurred in infancy, 35% in childhood, and 53% in adulthood (documented COD: 74%). For 261 shunted individuals, leading COD were neurological (21%) and pulmonary (17%), and leading MOD were infections (34%, including shunt infections: 4%) and non-infectious shunt malfunctions (14%). For 32 unshunted individuals, leading COD were pulmonary (34%) and cardiovascular (13%), and leading MOD were infections (38%) and non-infectious pulmonary (16%). COD and MOD varied by shunt status and age (p < = 0.04), not ambulation or birthyear (p > = 0.16). Urology-related deaths (urosepsis, renal failure, hematuria, bladder perforation/cancer: 10%) were more likely in females (p = 0.01), independent of age, shunt, or ambulatory status (p > = 0.40). COD/MOD were independent of bladder augmentation (p = >0.11). Unexplained deaths while asleep (4%) were independent of age, shunt status, and epilepsy (p >= 0.47). CONCLUSION: COD varied by shunt status. Leading MOD were infectious. Urology-related deaths (10%) were independent of shunt status; 26% of COD were unknown. Life-long multidisciplinary care and accurate mortality documentation are needed.",

keywords = "Myelomeningocele, cause of death, hydrocephalus, mortality, spinal dysraphism",

author = "Szymanski, {Konrad M.} and Adams, {Cyrus M.} and Alkawaldeh, {Mohammad Y.} and Austin, {Paul F.} and Bowman, {Robin M.} and Heidi Castillo and Jonathan Castillo and Chu, {David I.} and Estrada, {Carlos R.} and Michele Fascelli and Frimberger, {Dominic C.} and Gargollo, {Patricio C.} and Hamdan, {Dawud G.} and Hecht, {Sarah L.} and Betsy Hopson and Husmann, {Douglas A.} and Jacobs, {Micah A.} and MacNeily, {Andrew E.} and McLeod, {Daryl J.} and Metcalfe, {Peter D.} and Theresa Meyer and Rosalia Misseri and Joseph O'Neil and Rensing, {Adam J.} and Routh, {Jonathan C.} and Rove, {Kyle O.} and Sawin, {Kathleen J.} and Schlomer, {Bruce J.} and Isaac Shamblin and Sherlock, {Rebecca L.} and Gennady Slobodov and Jennifer Stout and Tanaka, {Stacy T.} and Weiss, {Dana A.} and Wiener, {John S.} and Wood, {Hadley M.} and Yerkes, {Elizabeth B.} and Jeffrey Blount",

note = "Publisher Copyright: {\textcopyright} 2023 - The authors. Published by IOS Press.",

year = "2023",

month = dec,

day = "26",

doi = "10.3233/PRM-220086",

language = "English (US)",

volume = "16",

pages = "605--619",

journal = "Journal of Pediatric Rehabilitation Medicine",

issn = "1874-5393",

publisher = "IOS Press",

number = "4",

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TY - JOUR

T1 - Causes of death among people with myelomeningocele

T2 - A multi-institutional 47-year retrospective study

AU - Szymanski, Konrad M.

AU - Adams, Cyrus M.

AU - Alkawaldeh, Mohammad Y.

AU - Austin, Paul F.

AU - Bowman, Robin M.

AU - Castillo, Heidi

AU - Castillo, Jonathan

AU - Chu, David I.

AU - Estrada, Carlos R.

AU - Fascelli, Michele

AU - Frimberger, Dominic C.

AU - Gargollo, Patricio C.

AU - Hamdan, Dawud G.

AU - Hecht, Sarah L.

AU - Hopson, Betsy

AU - Husmann, Douglas A.

AU - Jacobs, Micah A.

AU - MacNeily, Andrew E.

AU - McLeod, Daryl J.

AU - Metcalfe, Peter D.

AU - Meyer, Theresa

AU - Misseri, Rosalia

AU - O'Neil, Joseph

AU - Rensing, Adam J.

AU - Routh, Jonathan C.

AU - Rove, Kyle O.

AU - Sawin, Kathleen J.

AU - Schlomer, Bruce J.

AU - Shamblin, Isaac

AU - Sherlock, Rebecca L.

AU - Slobodov, Gennady

AU - Stout, Jennifer

AU - Tanaka, Stacy T.

AU - Weiss, Dana A.

AU - Wiener, John S.

AU - Wood, Hadley M.

AU - Yerkes, Elizabeth B.

AU - Blount, Jeffrey

PY - 2023/12/26

Y1 - 2023/12/26

N2 - PURPOSE: This study aimed to analyze organ system-based causes and non-organ system-based mechanisms of death (COD, MOD) in people with myelomeningocele (MMC), comparing urological to other COD. METHODS: A retrospective review was performed of 16 institutions in Canada/United States of non-random convenience sample of people with MMC (born > = 1972) using non-parametric statistics. RESULTS: Of 293 deaths (89% shunted hydrocephalus), 12% occurred in infancy, 35% in childhood, and 53% in adulthood (documented COD: 74%). For 261 shunted individuals, leading COD were neurological (21%) and pulmonary (17%), and leading MOD were infections (34%, including shunt infections: 4%) and non-infectious shunt malfunctions (14%). For 32 unshunted individuals, leading COD were pulmonary (34%) and cardiovascular (13%), and leading MOD were infections (38%) and non-infectious pulmonary (16%). COD and MOD varied by shunt status and age (p < = 0.04), not ambulation or birthyear (p > = 0.16). Urology-related deaths (urosepsis, renal failure, hematuria, bladder perforation/cancer: 10%) were more likely in females (p = 0.01), independent of age, shunt, or ambulatory status (p > = 0.40). COD/MOD were independent of bladder augmentation (p = >0.11). Unexplained deaths while asleep (4%) were independent of age, shunt status, and epilepsy (p >= 0.47). CONCLUSION: COD varied by shunt status. Leading MOD were infectious. Urology-related deaths (10%) were independent of shunt status; 26% of COD were unknown. Life-long multidisciplinary care and accurate mortality documentation are needed.

AB - PURPOSE: This study aimed to analyze organ system-based causes and non-organ system-based mechanisms of death (COD, MOD) in people with myelomeningocele (MMC), comparing urological to other COD. METHODS: A retrospective review was performed of 16 institutions in Canada/United States of non-random convenience sample of people with MMC (born > = 1972) using non-parametric statistics. RESULTS: Of 293 deaths (89% shunted hydrocephalus), 12% occurred in infancy, 35% in childhood, and 53% in adulthood (documented COD: 74%). For 261 shunted individuals, leading COD were neurological (21%) and pulmonary (17%), and leading MOD were infections (34%, including shunt infections: 4%) and non-infectious shunt malfunctions (14%). For 32 unshunted individuals, leading COD were pulmonary (34%) and cardiovascular (13%), and leading MOD were infections (38%) and non-infectious pulmonary (16%). COD and MOD varied by shunt status and age (p < = 0.04), not ambulation or birthyear (p > = 0.16). Urology-related deaths (urosepsis, renal failure, hematuria, bladder perforation/cancer: 10%) were more likely in females (p = 0.01), independent of age, shunt, or ambulatory status (p > = 0.40). COD/MOD were independent of bladder augmentation (p = >0.11). Unexplained deaths while asleep (4%) were independent of age, shunt status, and epilepsy (p >= 0.47). CONCLUSION: COD varied by shunt status. Leading MOD were infectious. Urology-related deaths (10%) were independent of shunt status; 26% of COD were unknown. Life-long multidisciplinary care and accurate mortality documentation are needed.

KW - Myelomeningocele

KW - cause of death

KW - hydrocephalus

KW - mortality

KW - spinal dysraphism

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DO - 10.3233/PRM-220086

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AN - SCOPUS:85181396099

SN - 1874-5393

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JO - Journal of Pediatric Rehabilitation Medicine

JF - Journal of Pediatric Rehabilitation Medicine

IS - 4

ER -

Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study

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