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• W1997194908 abstract "A 7-month-old infant presented with cough and fever. Platelet count was 50 000 × 103/mm3. Chest radiograph demonstrated diffuse nodular densities in both lungs. Computed tomography (CT) demonstrated innumerable, subcentimeter pulmonary nodules. Bronchoalveolar lavage was positive for Streptococcus pneumoniae, which was treated with antibiotics. Symptoms recurred after initial antibiotics were administered. Follow-up CT at 12 months of age showed interval increase in the size of pulmonary nodules (Figure 1; available at www.jpeds.com). Lytic lesions in the manubrium and vertebrae were also noted.Another infant presented at 10 months of age with 3 months of cough and hemoptysis. Complete blood count revealed hemoglobin 6.3 g/dL, hematocrit 22%, and platelet count 32 000 × 103/mm3. CT showed multiple, bilateral nodular opacities and ground-glass opacities. Bronchoscopy demonstrated blood in the trachea and bronchi; lavage cytology was compatible with pulmonary hemorrhage.Histopathologic features of lung biopsies were consistent with cutaneovisceral angiomatosis with thrombocytopenia (CAT) in both cases (Figure 2).CAT, also known as multifocal lymphangioendotheliomatosis with thrombocytopenia, is a vascular disorder affecting various organs and associated with variable thrombocytopenia. The cutaneous lesions, typically involving the trunk and extremities, are multiple, red-brown to blue macules, papules, and plaques ranging in diameter from millimeters to several centimeters.1North P.E. Kahn T. Cordisco M.R. Dadras S.S. Detmar M. Frieden I.J. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a newly recognized clinicopathological entity.Arch Dermatol. 2004; 140: 599-606Crossref PubMed Scopus (90) Google Scholar, 2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google Scholar CAT may also affect the gastrointestinal tract, lungs, bone, liver, spleen, synovia, and skeletal muscles.1North P.E. Kahn T. Cordisco M.R. Dadras S.S. Detmar M. Frieden I.J. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a newly recognized clinicopathological entity.Arch Dermatol. 2004; 140: 599-606Crossref PubMed Scopus (90) Google Scholar, 2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google Scholar, 3Piggott K.D. Riedel P.A. Baron H.I. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a rare cause of gastrointestinal bleeding in the newborn period.Pediatrics. 2006; 117: 810-813Crossref Scopus (22) Google Scholar Pulmonary involvement typically manifests as pulmonary nodules with pulmonary hemorrhage within the first 2 years of life.2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google ScholarAlthough cutaneous manifestations of CAT are present in the majority of patients, a subgroup of infants may present without cutaneous involvement. In the absence of known pulmonary disease, the association of extensive, multinodular, hemorrhagic pulmonary opacities and thrombocytopenia should raise the suspicion of CAT, considering biopsy is prudent to establish the diagnosis. A 7-month-old infant presented with cough and fever. Platelet count was 50 000 × 103/mm3. Chest radiograph demonstrated diffuse nodular densities in both lungs. Computed tomography (CT) demonstrated innumerable, subcentimeter pulmonary nodules. Bronchoalveolar lavage was positive for Streptococcus pneumoniae, which was treated with antibiotics. Symptoms recurred after initial antibiotics were administered. Follow-up CT at 12 months of age showed interval increase in the size of pulmonary nodules (Figure 1; available at www.jpeds.com). Lytic lesions in the manubrium and vertebrae were also noted. Another infant presented at 10 months of age with 3 months of cough and hemoptysis. Complete blood count revealed hemoglobin 6.3 g/dL, hematocrit 22%, and platelet count 32 000 × 103/mm3. CT showed multiple, bilateral nodular opacities and ground-glass opacities. Bronchoscopy demonstrated blood in the trachea and bronchi; lavage cytology was compatible with pulmonary hemorrhage. Histopathologic features of lung biopsies were consistent with cutaneovisceral angiomatosis with thrombocytopenia (CAT) in both cases (Figure 2). CAT, also known as multifocal lymphangioendotheliomatosis with thrombocytopenia, is a vascular disorder affecting various organs and associated with variable thrombocytopenia. The cutaneous lesions, typically involving the trunk and extremities, are multiple, red-brown to blue macules, papules, and plaques ranging in diameter from millimeters to several centimeters.1North P.E. Kahn T. Cordisco M.R. Dadras S.S. Detmar M. Frieden I.J. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a newly recognized clinicopathological entity.Arch Dermatol. 2004; 140: 599-606Crossref PubMed Scopus (90) Google Scholar, 2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google Scholar CAT may also affect the gastrointestinal tract, lungs, bone, liver, spleen, synovia, and skeletal muscles.1North P.E. Kahn T. Cordisco M.R. Dadras S.S. Detmar M. Frieden I.J. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a newly recognized clinicopathological entity.Arch Dermatol. 2004; 140: 599-606Crossref PubMed Scopus (90) Google Scholar, 2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google Scholar, 3Piggott K.D. Riedel P.A. Baron H.I. Multifocal lymphangioendotheliomatosis with thrombocytopenia: a rare cause of gastrointestinal bleeding in the newborn period.Pediatrics. 2006; 117: 810-813Crossref Scopus (22) Google Scholar Pulmonary involvement typically manifests as pulmonary nodules with pulmonary hemorrhage within the first 2 years of life.2Prasad V. Fishman S.J. Mulliken J.B. Fox V.L. Liang M.G. Klement G. et al.Cutaneovisceral angiomatosis with thrombocytopenia.Pediatr Dev Pathol. 2005; 8: 407-419Crossref PubMed Scopus (51) Google Scholar Although cutaneous manifestations of CAT are present in the majority of patients, a subgroup of infants may present without cutaneous involvement. In the absence of known pulmonary disease, the association of extensive, multinodular, hemorrhagic pulmonary opacities and thrombocytopenia should raise the suspicion of CAT, considering biopsy is prudent to establish the diagnosis. Appendix" @default.
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• W1997194908 title "Cutaneovisceral Angiomatosis with Thrombocytopenia without Cutaneous Involvement" @default.
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