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Lymphangiomatosis involving the pulmonary and extrapulmonary lymph nodes and surrounding soft tissue: A rare case report.

Abstract Diffuse pulmonary lymphangiomatosis (DPL) mainly affects the lung and pleura. There are very few pathological reports of lung damage accompanied by diffuse involvement of the extrapulmonary lymph nodes and surrounding soft tissue. The clinicopathological significance of coexistence of pulmonary and extrapulmonary lesions is unknown.
PMID
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Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start




PMID- 29245295
OWN - NLM
STAT- MEDLINE
DCOM- 20171226
LR  - 20171226
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 49
DP  - 2017 Dec
TI  - Lymphangiomatosis involving the pulmonary and extrapulmonary lymph nodes and
      surrounding soft tissue: A rare case report.
PG  - e9032
LID - 10.1097/MD.0000000000009032 [doi]
AB  - BACKGROUND: Diffuse pulmonary lymphangiomatosis (DPL) mainly affects the lung and
      pleura. There are very few pathological reports of lung damage accompanied by
      diffuse involvement of the extrapulmonary lymph nodes and surrounding soft
      tissue. The clinicopathological significance of coexistence of pulmonary and
      extrapulmonary lesions is unknown. METHODS: Here, we report a 16-year-old male
      patient. The pathological specimens of the supraclavicular lymph node and soft
      tissue together with the lung biopsy were analyzed by pathological observation
      and immunohistochemical staining. Literatures were reviewed and clinical and
      imaging findings were discussed. RESULTS: The patient presented with coughing and
      expectoration for 1 year and intermittent hemoptysis for 4 months. Ultrasound
      revealed swollen lymph nodes in bilateral neck, left armpit, and pubic symphysis.
      Chest CT scan showed diffuse grid and linear shadows, bilateral pleural
      thickening, and nodule formation. Multiple enlarged lymph nodes were mainly
      investigated in bilateral hilar, mediastinal, para-aortic, lesser curvature, and 
      retroperitoneal. Supraclavicular lymph node biopsy confirmed the lymphatic
      hyperplasia and expansion in the capsule and surrounding soft tissue. The
      thoracoscopic examination found bloody chylothorax on the left chest. And lung
      biopsy showed the lymphatic vessel hyperplasia and expansion on the pleura and
      adjacent lung tissue. Immunohistochemical stains showed that the lymphatic
      endothelial cells were positive for D2-40 and CD31. Lymphangiomatosis involving
      the pulmonary and extrapulmonary lymph nodes and surrounding soft tissue was
      diagnosed based on the aforementioned histological findings. CONCLUSION:
      Lymphangiomatosis of superficial lymph node mainly involves the capsule of lymph 
      nodes and its surrounding soft tissue. The information obtained from the lymph
      node biopsy can prompt and assist the diagnosis of DPL.
FAU - Fang, Xuhua
AU  - Fang X
AD  - aDepartment of ENT, Children's Medical Center, Shanghai Jiao Tong University,
      ShanghaibDepartment of Pathology, Shanghai Tongji Hospital, Tongji Hospital
      Affiliated to Tongji University, ShanghaicDepartment of Pathology, Chongqing
      Medical University, ChongqingdDepartment of Pathology, Shanghai Pulmonary
      Hospital, Tongji University School of Medicine, Shanghai, China.
FAU - Huang, Ziling
AU  - Huang Z
FAU - Zeng, Yu
AU  - Zeng Y
FAU - Zhu, Xuyou
AU  - Zhu X
FAU - Wang, Siqi
AU  - Wang S
FAU - Yu, Xiaoting
AU  - Yu X
FAU - Li, Xian
AU  - Li X
FAU - Wu, Chunyan
AU  - Wu C
FAU - Yi, Xianghua
AU  - Yi X
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - Lymphangiectasia, pulmonary, congenital
SB  - AIM
SB  - IM
MH  - Adolescent
MH  - Biopsy
MH  - Humans
MH  - Lung Diseases/*congenital/pathology
MH  - Lymph Nodes/*pathology
MH  - Lymphangiectasis/*congenital/pathology
MH  - Male
MH  - Neck/pathology
MH  - Thorax/pathology
PMC - PMC5728910
EDAT- 2017/12/17 06:00
MHDA- 2017/12/27 06:00
CRDT- 2017/12/17 06:00
PHST- 2017/12/17 06:00 [entrez]
PHST- 2017/12/17 06:00 [pubmed]
PHST- 2017/12/27 06:00 [medline]
AID - 10.1097/MD.0000000000009032 [doi]
AID - 00005792-201712080-00084 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Dec;96(49):e9032. doi: 10.1097/MD.0000000000009032.