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Bean's syndrome in children: about two cases.

Abstract Diffuse angiomatosis or Bean's syndrome is a rare disease characterized by venous malformations mainly involving the skin and the digestive tract which can result in hemorrhage of variable severity. This study reports the case of two children aged 5 and 9 and a half years respectively with diffuse angiomatosis who had been treated in the Department of Emergency Paediatric Surgery over the years. The diagnosis was based on rectal bleeding and/or melenas causing severe anemia requiring regular transfusions in both patients as well as skin angiomas occurrence at the level of the limbs. Radiological evaluation showed the presence of multiple lesions at the level of the jejunum and ileum consistent with small intestinal angiomatosis in the child aged 9 and a half years. It didn't show abdominal lesions in the child aged 5 years. The two patients were admitted to the operating block. Angiomas were surgically individualized. Some of them were actively bleeding. Enterotomy was performed. Postoperative sequelae was marked by the stop of the bleedings. This study aims to update the current understanding of this rare pathology as well as the benefit of surgical treatment in controlling the complications caused by this pathology and in reducing the frequency of transfusions.
PMID
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Authors

Mayor MeshTerms
Keywords

Angiomatosis

Bean's syndrome

surgery

Journal Title the pan african medical journal
Publication Year Start




PMID- 29515720
OWN - NLM
STAT- MEDLINE
DCOM- 20180313
LR  - 20180313
IS  - 1937-8688 (Electronic)
VI  - 28
DP  - 2017
TI  - [Bean's syndrome in children: about two cases].
PG  - 102
LID - 10.11604/pamj.2017.28.102.11109 [doi]
AB  - Diffuse angiomatosis or Bean's syndrome is a rare disease characterized by venous
      malformations mainly involving the skin and the digestive tract which can result 
      in hemorrhage of variable severity. This study reports the case of two children
      aged 5 and 9 and a half years respectively with diffuse angiomatosis who had been
      treated in the Department of Emergency Paediatric Surgery over the years. The
      diagnosis was based on rectal bleeding and/or melenas causing severe anemia
      requiring regular transfusions in both patients as well as skin angiomas
      occurrence at the level of the limbs. Radiological evaluation showed the presence
      of multiple lesions at the level of the jejunum and ileum consistent with small
      intestinal angiomatosis in the child aged 9 and a half years. It didn't show
      abdominal lesions in the child aged 5 years. The two patients were admitted to
      the operating block. Angiomas were surgically individualized. Some of them were
      actively bleeding. Enterotomy was performed. Postoperative sequelae was marked by
      the stop of the bleedings. This study aims to update the current understanding of
      this rare pathology as well as the benefit of surgical treatment in controlling
      the complications caused by this pathology and in reducing the frequency of
      transfusions.
FAU - El Bakkaly, Achraf
AU  - El Bakkaly A
AD  - Service des Urgences Chirurgicales Pediatriques, CHU Ibn Sina, Faculte de
      Medecine Mohammed V, Rabat, Maroc.
FAU - Ettayebi, Fouad
AU  - Ettayebi F
AD  - Service des Urgences Chirurgicales Pediatriques, CHU Ibn Sina, Faculte de
      Medecine Mohammed V, Rabat, Maroc.
FAU - Oubeja, Houda
AU  - Oubeja H
AD  - Service des Urgences Chirurgicales Pediatriques, CHU Ibn Sina, Faculte de
      Medecine Mohammed V, Rabat, Maroc.
FAU - Erraji, Mounir
AU  - Erraji M
AD  - Service des Urgences Chirurgicales Pediatriques, CHU Ibn Sina, Faculte de
      Medecine Mohammed V, Rabat, Maroc.
FAU - Zerhouni, Hicham
AU  - Zerhouni H
AD  - Service des Urgences Chirurgicales Pediatriques, CHU Ibn Sina, Faculte de
      Medecine Mohammed V, Rabat, Maroc.
LA  - fre
PT  - Case Reports
PT  - Journal Article
TT  - Syndrome de Bean chez l'enfant: a propos de deux cas.
DEP - 20171004
PL  - Uganda
TA  - Pan Afr Med J
JT  - The Pan African medical journal
JID - 101517926
RN  - Blue rubber bleb nevus syndrome
SB  - IM
MH  - Anemia/*etiology
MH  - Angiomatosis/*etiology
MH  - Child
MH  - Child, Preschool
MH  - Gastrointestinal Neoplasms/*diagnosis/surgery
MH  - Humans
MH  - Nevus, Blue/*diagnosis/surgery
MH  - Skin Neoplasms/*diagnosis/surgery
PMC - PMC5837144
OTO - NOTNLM
OT  - Angiomatosis
OT  - Bean's syndrome
OT  - surgery
EDAT- 2018/03/09 06:00
MHDA- 2018/03/14 06:00
CRDT- 2018/03/09 06:00
PHST- 2016/11/06 00:00 [received]
PHST- 2017/08/24 00:00 [accepted]
PHST- 2018/03/09 06:00 [entrez]
PHST- 2018/03/09 06:00 [pubmed]
PHST- 2018/03/14 06:00 [medline]
AID - 10.11604/pamj.2017.28.102.11109 [doi]
AID - PAMJ-28-102 [pii]
PST - epublish
SO  - Pan Afr Med J. 2017 Oct 4;28:102. doi: 10.11604/pamj.2017.28.102.11109.
      eCollection 2017.