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Lymphangiomyomatosis — Clinical Guidelines

Overview

Lymphangiomyomatosis (LAM) is a rare, progressive disorder characterized by abnormal proliferation of lymphatic vessels, leading to cystic changes primarily in the lungs but potentially involving other organs such as the spleen and mediastinum. 1 3

Diagnosis

Clinical Presentation: May include progressive dyspnea, cough, hemoptysis, and abdominal fullness. 1 3

Imaging: CT and MRI reveal cystic lesions, splenomegaly, and involvement of multiple organ systems. 1 3

Special Manifestations: Disseminated intravascular coagulation (DIC) can occur in pediatric cases, alongside bone lesions. 2

Diagnostic Criteria: Radiologic findings are crucial, often supplemented by histopathological confirmation in surgical specimens. 3

Management

Surgical Intervention: Considered for severe cases, particularly thoracic multiorgan involvement, such as pulmonary lymphangiectomy. 3

Medical Management: Total parenteral nutrition, fat-free diets, and octreotide may be used for symptom control and complications like chylothorax. 3

Splenectomy: Indicated in cases of massive splenomegaly with cystic lesions causing significant compression or complications. 1

Special Populations

Pediatrics: May present with unusual manifestations like DIC and bone lesions. 2

Comorbidities: Portal hypertension and associated varices can coexist with splenic involvement. 1

Key Recommendations

Surgical Evaluation for Multiorgan Involvement: Consider surgical options like pulmonary lymphangiectomy in patients with severe thoracic multiorgan lymphangiomatosis, despite high-risk preoperative assessments. (Evidence: Moderate 3)

Aggressive Management of Complications: Employ total parenteral nutrition and octreotide for managing complications such as chylothorax in patients with thoracic lymphangiomatosis. (Evidence: Weak 3)

Surgical Intervention for Severe Splenomegaly: Perform splenectomy in cases of symptomatic, massive splenomegaly due to cystic lymphangiomatosis. (Evidence: Expert opinion 1)

References

1 Hsu WF, Yu CJ, Yeh CC, Huang WH, Peng CY. Splenic cystic lymphangiomatosis in association with omental varices and portal hypertension: A case report. Medicine 2018. link 2 Prieur O, Damry N, Heijmans C, Christophe C. Unusual clinical manifestation of lymphangiomatosis. JBR-BTR : organe de la Societe royale belge de radiologie (SRBR) = orgaan van de Koninklijke Belgische Vereniging voor Radiologie (KBVR) 2011. link 3 Steinacher I, Lamprecht B, Lobendanz M, Zoller H, Dartevelle P, Fadel E et al.. Successful surgical treatment of thoracic multiorgan lymphangiomatosis. Wiener klinische Wochenschrift 2009. link

Lymphangiomyomatosis