Dermatology: Practical and Conceptual


1

Intralymphatic Histiocytosis
Emilio Garcia-Mouronte1, Borja Diaz-Guimaraens1, Emilio de Dios Berna-Rico1, 

Miguel Dominguez-Santas1

1 Dermatology Department, Ramon y Cajal University Hospital, Madrid, Spain 

Citation: Garcia-Mouronte E, Diaz-Guimaraens B, Berna-Rico E, Dominguez-Santas M. Intralymphatic histiocytosis. 
Dermatol Pract Concept. 2022;12(2):e2022067. DOI: https://doi.org/10.5826/dpc.1202a67

Accepted: September 6, 2021; Published: April 2022

Copyright: ©2022 Iorizzo et al. This is an open-access article distributed under the terms of the Creative Commons  
Attribution-NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted 
noncommercial use, distribution, and reproduction in any medium, provided the original authors and source are credited.

Funding: None.

Competing interests: None.

Authorship: All authors have contributed significantly to this publication.

Corresponding author: Emilio Garcia-Mouronte, MD. Dermatology Department, Ramon y Cajal University Hospital. Carretera Colmenar 
Viejo km 9.100, 28034 Madrid, Spain. E-mail: emilio.garcia.mouronte@gmail.com

Case Presentation

An 87-year-old man presented to our Dermatology depart-

ment with a history of 1-year-long asymptomatic erythem-

atous plaques on his left shoulder. He referred he had been 

previously diagnosed from severe osteoarthritis on this loca-

tion. Moreover, he denied taking regular medication, expo-

sure to heat or suffering from any other medical condition. 

Dermatological examination showed 2 mm wide serpig-

inous erythematous plaques with a vascular anatomic dis-

tribution on the anterior side of his left shoulder (Figure 1). 

Neither cervical nor axillary adenopathies were detected. 

After taking a punch-biopsy, a diagnosis of intralymphatic 

histiocytosis was established. 

Teaching point

Intralymphatic histiocytosis is a benign proliferation of 

histiocytes within lymphatic vessels [1]. Although its etio-

pathogenesis has not yet been clearly elucidated, it has been 

associated to several chronic inflammatory and degenerative 

disorders, such as osteoarthritis [2]. Thus, Dermatologists 

should take into account this entity whenever they are facing 

serpiginous erythematous plaques with a vascular distribu-

tion on a persistent swollen joint. 

Image Letter | Dermatol Pract Concept. 2022;12(2):e2022067

Figure 1. 2 mm wide serpiginous erythematous plaques with a 

vascular distribution on the left shoulder.



2 Research Letter | Dermatol Pract Concept. 2022;12(2):e2022067

References

1. Takiwaki H, Adachi A, Kohno H, Ogawa Y. Intravascular or in-

tralymphatic histiocytosis associated with rheumatoid arthritis:

A report of 4 cases. J Am Acad Dermatol. 2004;50(4):585–590.

DOI: 10.1016/j.jaad.2003.09.025. PMID: 15034508.

2. Korman JB, Burgin S, Tahan SR. Intralymphatic histiocyto-

sis in association with severe osteoarthritis of the shoulder.

J Am Acad Dermatol. 2013;69(6):e314–e315. DOI: 10.1016/j.

jaad.2013.08.020. PMID: 24238195.