Anorectal malformation, urethral duplication, occult spinal dysraphism (ARM-UD-OSD): a challenging uncommon association.


Journal

Pediatric surgery international
ISSN: 1437-9813
Titre abrégé: Pediatr Surg Int
Pays: Germany
ID NLM: 8609169

Informations de publication

Date de publication:
Oct 2022
Historique:
accepted: 14 07 2022
pubmed: 27 7 2022
medline: 14 9 2022
entrez: 26 7 2022
Statut: ppublish

Résumé

Urethral duplication (UD) is a rare malformation, which can be associated with other anomalies, like anorectal malformations (ARM). ARM has been described with occult spinal dysraphism (OSD). No ARM-UD-OSD combination has been reported. To share our experience and to discuss the management of ARM-UD-OSD association. We retrospectively reviewed records of five boys with UD. Four of these had ARM-UD-OSD association. ARM was the first diagnosis in all; OSD and UD was detected during screening for associated malformation. All patients underwent ARM correction, 3 after colostomy. All reached fecal continence, 3 are performing bowel management. Three patients underwent UD surgical correction. Because of symptoms' worsening, 2 children had detethering surgery. At a mean follow-up of 9.5 years, all patients have normal renal function, 3 are on clean intermittent catheterization (CIC) for neurogenic bladder (1 has a cystostomy, another one an appendicostomy). UD and OSD should be considered in patients with ARM. Children with these conditions associated must be centralized in a third-level Center and management carefully planned; in particular, urethral reconstruction should be weighed, considering CIC could be required. Suspicion of neurogenic bladder must be present in OSD patient.

Identifiants

pubmed: 35882671
doi: 10.1007/s00383-022-05186-z
pii: 10.1007/s00383-022-05186-z
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1487-1494

Informations de copyright

© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

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Auteurs

Federica Lena (F)

Division of Neuro-Urology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.
University of Genoa, DINOGMI, Largo Paolo Daneo 3, 16132, Genoa, Italy.

Chiara Pellegrino (C)

Division of Neuro-Urology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy. chiara.pellegrino@opbg.net.

Antonio Maria Zaccara (AM)

Division of Neuro-Urology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Maria Luisa Capitanucci (ML)

Division of Neuro-Urology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Giacomo Esposito (G)

Department of Neuroscience, Neurosurgery Unit, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Barbara Daniela Iacobelli (BD)

Medical and Surgical Department of the Fetus-Newborn-Infant, Newborn Surgery Unit, Bambino Gesu' Children's Hospital IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Daniela Longo (D)

Imaging Department, Neuroradiology Unit, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Tamara Caldaro (T)

Division of Digestive Surgery and Endoscopy, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Diletta Bruno (D)

Department of Neurorehabilitation, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Francesca Bevilacqua (F)

Department of Neuroscience, Unit of Clinical Psychology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Francesca Santato (F)

Department of Neuroscience, Unit of Clinical Psychology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Giulia Lucignani (G)

Imaging Department, Neuroradiology Unit, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Carlo Efisio Marras (CE)

Department of Neuroscience, Neurosurgery Unit, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Enrico Castelli (E)

Department of Neurorehabilitation, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Pietro Bagolan (P)

Medical and Surgical Department of the Fetus-Newborn-Infant, Newborn Surgery Unit, Bambino Gesu' Children's Hospital IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.
Department of Systems Medicine, University of Rome "Tor Vergata", Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

Giovanni Mosiello (G)

Division of Neuro-Urology, Bambino Gesu' Children's Hospital, IRCCS, Piazza di Sant'Onofrio 4, 00165, Rome, Italy.

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