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Spontaneous hemothorax: a rare complication of neurofibromatosis type 1.

Abstract Neurofibromatosis type 1 (NF1), also known as Von Recklinghausen's disease is an autosomal dominant genetic disorder. It is the most common of phacomatoses. Pulmonary complications have been rarely described in the literature. Vascular complications have been reported in 3.6% of patients. We here report the case of a 38-year old female patient, followed-up for neurofibromatosis type 1, admitted to the Emergency Department with hemorrhagic shock. Clinical examination showed several coffee-with-milk colored spots, many plexiform neurofibromas, left-sided pleural effusion syndrome. Pleural puncture objectified coagulable haemorrhagic fluid. The patient received transfusion and emergency chest drainage. Patient's assessment was completed by angioscanner which showed no pulmonary embolism or other associated lesions. Spontaneous hemothorax is a rare and severe complication of neurofibromatosis. It is probably due to vascular injury caused by this disease.
PMID
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Authors

Mayor MeshTerms
Keywords

Hemothorax

neurofibromatosis type 1

spontaneous

Journal Title the pan african medical journal
Publication Year Start




PMID- 29255555
OWN - NLM
STAT- MEDLINE
DCOM- 20171222
LR  - 20171222
IS  - 1937-8688 (Electronic)
VI  - 28
DP  - 2017
TI  - [Spontaneous hemothorax: a rare complication of neurofibromatosis type 1].
PG  - 85
LID - 10.11604/pamj.2017.28.85.13820 [doi]
AB  - Neurofibromatosis type 1 (NF1), also known as Von Recklinghausen's disease is an 
      autosomal dominant genetic disorder. It is the most common of phacomatoses.
      Pulmonary complications have been rarely described in the literature. Vascular
      complications have been reported in 3.6% of patients. We here report the case of 
      a 38-year old female patient, followed-up for neurofibromatosis type 1, admitted 
      to the Emergency Department with hemorrhagic shock. Clinical examination showed
      several coffee-with-milk colored spots, many plexiform neurofibromas, left-sided 
      pleural effusion syndrome. Pleural puncture objectified coagulable haemorrhagic
      fluid. The patient received transfusion and emergency chest drainage. Patient's
      assessment was completed by angioscanner which showed no pulmonary embolism or
      other associated lesions. Spontaneous hemothorax is a rare and severe
      complication of neurofibromatosis. It is probably due to vascular injury caused
      by this disease.
FAU - Fdil, Soumia
AU  - Fdil S
AD  - Service de Pneumologie, Hopital Moulay Youssef, Faculte de Medecine et de
      Pharmacie, Universite Mohammed V, CHU Ibn Sina, 10000 Rabat, Maro.
FAU - Bouchikhi, Saad
AU  - Bouchikhi S
AD  - Service de Pneumologie, Hopital Moulay Youssef, Faculte de Medecine et de
      Pharmacie, Universite Mohammed V, CHU Ibn Sina, 10000 Rabat, Maro.
FAU - Bourkadi, Jamal-Eddine
AU  - Bourkadi JE
AD  - Service de Pneumologie, Hopital Moulay Youssef, Faculte de Medecine et de
      Pharmacie, Universite Mohammed V, CHU Ibn Sina, 10000 Rabat, Maro.
LA  - fre
PT  - Case Reports
PT  - Journal Article
TT  - Hemothorax spontane: complication rare de la neurofibromatose type 1.
DEP - 20170927
PL  - Uganda
TA  - Pan Afr Med J
JT  - The Pan African medical journal
JID - 101517926
SB  - IM
MH  - Adult
MH  - Blood Transfusion
MH  - Female
MH  - Hemothorax/*etiology/therapy
MH  - Humans
MH  - Neurofibroma, Plexiform/etiology
MH  - Neurofibromatosis 1/*complications
MH  - Pleural Effusion/*etiology/therapy
MH  - Shock, Hemorrhagic/*etiology/therapy
PMC - PMC5724956
OTO - NOTNLM
OT  - Hemothorax
OT  - neurofibromatosis type 1
OT  - spontaneous
EDAT- 2017/12/20 06:00
MHDA- 2017/12/23 06:00
CRDT- 2017/12/20 06:00
PHST- 2017/09/06 00:00 [received]
PHST- 2017/09/12 00:00 [accepted]
PHST- 2017/12/20 06:00 [entrez]
PHST- 2017/12/20 06:00 [pubmed]
PHST- 2017/12/23 06:00 [medline]
AID - 10.11604/pamj.2017.28.85.13820 [doi]
AID - PAMJ-28-85 [pii]
PST - epublish
SO  - Pan Afr Med J. 2017 Sep 27;28:85. doi: 10.11604/pamj.2017.28.85.13820.
      eCollection 2017.