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Unusual macrocystic lymphatic malformation in an adult patient.

Abstract Macrocystic lymphatic malformations (MLMs) constitute a circumscribed variation in deep lymphangiomas. They are characterized by a rare occurrence in adults, by their rapid expansion and by their frequent occurrence at the level of the cervicofacial region (75%) and axillary region (20%). They rarely occur in lower limbs. The diagnosis of MLMs is primarily based on clinical examination. Ultrasound has a valuable role in positive diagnosis and sometimes in differential diagnosis. MRI has a role in the diagnosis and the assessment of tumor extent, thus making a valuable contribution to surgery. Therapeutically, MLMs are mainly treated by sclerotherapy; surgery is rarely indicated. We report the case of 26-year old man with a one-year history of huge painless and rapidly evolving subcutaneous swelling in the right thigh. Clinical examination showed circumferential compressible non-beating infiltrated mass, surmounted by vegetative and ulcerated lesions. The remainder of the clinical examination showed the presence of inguinal ipsilateral lymphadenopathy, with mobility limitation of the right knee. Angio-MRI objectified diffuse heterogeneous gadolinium-enhancing cutaneous, subcutaneous thickening of the right thigh with perimuscolar aponeurotic involvement. Lymphoscintigraphy showed asymmetry of the accumultion of radiopharmaceutical kinetics that was reduced in the right lower limb in favor of an overload of the lymphatic system at rest. Given the clinico-radiological data, the diagnosis of MLM was retained. Given the seat of the lymphangioma and its extent, after consultation with the surgeons there was no indication for surgery. Immobilization with a removable bandage was indicated for our patient.
PMID
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Authors

Mayor MeshTerms
Keywords

Lymphatic malformation

lymphangioma

macrocystic lymphangioma

Journal Title the pan african medical journal
Publication Year Start




PMID- 29515746
OWN - NLM
STAT- MEDLINE
DCOM- 20180313
LR  - 20180313
IS  - 1937-8688 (Electronic)
VI  - 28
DP  - 2017
TI  - [Unusual macrocystic lymphatic malformation in an adult patient].
PG  - 128
LID - 10.11604/pamj.2017.28.128.13472 [doi]
AB  - Macrocystic lymphatic malformations (MLMs) constitute a circumscribed variation
      in deep lymphangiomas. They are characterized by a rare occurrence in adults, by 
      their rapid expansion and by their frequent occurrence at the level of the
      cervicofacial region (75%) and axillary region (20%). They rarely occur in lower 
      limbs. The diagnosis of MLMs is primarily based on clinical examination.
      Ultrasound has a valuable role in positive diagnosis and sometimes in
      differential diagnosis. MRI has a role in the diagnosis and the assessment of
      tumor extent, thus making a valuable contribution to surgery. Therapeutically,
      MLMs are mainly treated by sclerotherapy; surgery is rarely indicated. We report 
      the case of 26-year old man with a one-year history of huge painless and rapidly 
      evolving subcutaneous swelling in the right thigh. Clinical examination showed
      circumferential compressible non-beating infiltrated mass, surmounted by
      vegetative and ulcerated lesions. The remainder of the clinical examination
      showed the presence of inguinal ipsilateral lymphadenopathy, with mobility
      limitation of the right knee. Angio-MRI objectified diffuse heterogeneous
      gadolinium-enhancing cutaneous, subcutaneous thickening of the right thigh with
      perimuscolar aponeurotic involvement. Lymphoscintigraphy showed asymmetry of the 
      accumultion of radiopharmaceutical kinetics that was reduced in the right lower
      limb in favor of an overload of the lymphatic system at rest. Given the
      clinico-radiological data, the diagnosis of MLM was retained. Given the seat of
      the lymphangioma and its extent, after consultation with the surgeons there was
      no indication for surgery. Immobilization with a removable bandage was indicated 
      for our patient.
FAU - Ramid, AHind
AU  - Ramid A
AD  - Service de Dermatologie-Venereologie, CHU Ibn Rochd, Universite Hassan II,
      Casablanca, Maroc.
FAU - Hali, Fouzia
AU  - Hali F
AD  - Service de Dermatologie-Venereologie, CHU Ibn Rochd, Universite Hassan II,
      Casablanca, Maroc.
LA  - fre
PT  - Case Reports
PT  - Journal Article
TT  - Malformation lymphatique macrokystique atypique chez un adulte.
DEP - 20171010
PL  - Uganda
TA  - Pan Afr Med J
JT  - The Pan African medical journal
JID - 101517926
SB  - IM
MH  - Adult
MH  - Diagnosis, Differential
MH  - Humans
MH  - Lymphangioma, Cystic/*diagnosis/pathology/therapy
MH  - Lymphatic Abnormalities/*diagnosis/pathology/therapy
MH  - Magnetic Resonance Imaging
MH  - Male
MH  - Sclerotherapy/*methods
MH  - Ultrasonography
PMC - PMC5837141
OTO - NOTNLM
OT  - Lymphatic malformation
OT  - lymphangioma
OT  - macrocystic lymphangioma
EDAT- 2018/03/09 06:00
MHDA- 2018/03/14 06:00
CRDT- 2018/03/09 06:00
PHST- 2017/07/27 00:00 [received]
PHST- 2017/09/05 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.2017.28.128.13472 [doi]
AID - PAMJ-28-128 [pii]
PST - epublish
SO  - Pan Afr Med J. 2017 Oct 10;28:128. doi: 10.11604/pamj.2017.28.128.13472.
      eCollection 2017.