Early-onset sarcoidosis mimicking refractory cutaneous histiocytosis

Shouichi Ohga, Kiyomi Ichino, Kazunori Urabe, Masataka Ishimura, Hidetoshi Takada, Ryuta Nishikomori, Masutaka Furue, Toshiro Hara

Research output: Contribution to journalArticle

3 Citations (Scopus)

Abstract

A 10-year-old female was diagnosed as having early-onset sarcoidosis (EOS) after a prolonged skin disease. A granuloma emerged on the face at age 2 and massive lesions extended to the rest of the body. Repeated biopsies indicated histiocytic proliferation. At age 7, fever, disseminated macular eruptions, and multinucleated giant cells in the bone marrow prompted vinblastine and prednisolone therapy. Five months after stopping therapy, hypercalcemic crisis occurred along with fever, cytopenias, and interferon-γ-nemia indicating a macrophage activation syndrome. A biopsy of nodules confirmed the diagnosis of sarcoidosis. The atypical EOS should be differentiated from histiocytosis.

Original languageEnglish
Pages (from-to)723-726
Number of pages4
JournalPediatric Blood and Cancer
Volume50
Issue number3
DOIs
Publication statusPublished - Mar 1 2008

Fingerprint

Histiocytosis
Macrophage Activation Syndrome
Fever
Biopsy
Skin
Vinblastine
Giant Cells
Sarcoidosis
Prednisolone
Granuloma
Skin Diseases
Interferons
Bone Marrow
Therapeutics
Early-Onset Sarcoidosis
Blau syndrome

All Science Journal Classification (ASJC) codes

  • Pediatrics, Perinatology, and Child Health
  • Hematology
  • Oncology

Cite this

Early-onset sarcoidosis mimicking refractory cutaneous histiocytosis. / Ohga, Shouichi; Ichino, Kiyomi; Urabe, Kazunori; Ishimura, Masataka; Takada, Hidetoshi; Nishikomori, Ryuta; Furue, Masutaka; Hara, Toshiro.

In: Pediatric Blood and Cancer, Vol. 50, No. 3, 01.03.2008, p. 723-726.

Research output: Contribution to journalArticle

Ohga, Shouichi ; Ichino, Kiyomi ; Urabe, Kazunori ; Ishimura, Masataka ; Takada, Hidetoshi ; Nishikomori, Ryuta ; Furue, Masutaka ; Hara, Toshiro. / Early-onset sarcoidosis mimicking refractory cutaneous histiocytosis. In: Pediatric Blood and Cancer. 2008 ; Vol. 50, No. 3. pp. 723-726.
@article{8084ec7790674530a36209282ba5b98d,
title = "Early-onset sarcoidosis mimicking refractory cutaneous histiocytosis",
abstract = "A 10-year-old female was diagnosed as having early-onset sarcoidosis (EOS) after a prolonged skin disease. A granuloma emerged on the face at age 2 and massive lesions extended to the rest of the body. Repeated biopsies indicated histiocytic proliferation. At age 7, fever, disseminated macular eruptions, and multinucleated giant cells in the bone marrow prompted vinblastine and prednisolone therapy. Five months after stopping therapy, hypercalcemic crisis occurred along with fever, cytopenias, and interferon-γ-nemia indicating a macrophage activation syndrome. A biopsy of nodules confirmed the diagnosis of sarcoidosis. The atypical EOS should be differentiated from histiocytosis.",
author = "Shouichi Ohga and Kiyomi Ichino and Kazunori Urabe and Masataka Ishimura and Hidetoshi Takada and Ryuta Nishikomori and Masutaka Furue and Toshiro Hara",
year = "2008",
month = "3",
day = "1",
doi = "10.1002/pbc.21173",
language = "English",
volume = "50",
pages = "723--726",
journal = "Pediatric Blood and Cancer",
issn = "1545-5009",
publisher = "Wiley-Liss Inc.",
number = "3",

}

TY - JOUR

T1 - Early-onset sarcoidosis mimicking refractory cutaneous histiocytosis

AU - Ohga, Shouichi

AU - Ichino, Kiyomi

AU - Urabe, Kazunori

AU - Ishimura, Masataka

AU - Takada, Hidetoshi

AU - Nishikomori, Ryuta

AU - Furue, Masutaka

AU - Hara, Toshiro

PY - 2008/3/1

Y1 - 2008/3/1

N2 - A 10-year-old female was diagnosed as having early-onset sarcoidosis (EOS) after a prolonged skin disease. A granuloma emerged on the face at age 2 and massive lesions extended to the rest of the body. Repeated biopsies indicated histiocytic proliferation. At age 7, fever, disseminated macular eruptions, and multinucleated giant cells in the bone marrow prompted vinblastine and prednisolone therapy. Five months after stopping therapy, hypercalcemic crisis occurred along with fever, cytopenias, and interferon-γ-nemia indicating a macrophage activation syndrome. A biopsy of nodules confirmed the diagnosis of sarcoidosis. The atypical EOS should be differentiated from histiocytosis.

AB - A 10-year-old female was diagnosed as having early-onset sarcoidosis (EOS) after a prolonged skin disease. A granuloma emerged on the face at age 2 and massive lesions extended to the rest of the body. Repeated biopsies indicated histiocytic proliferation. At age 7, fever, disseminated macular eruptions, and multinucleated giant cells in the bone marrow prompted vinblastine and prednisolone therapy. Five months after stopping therapy, hypercalcemic crisis occurred along with fever, cytopenias, and interferon-γ-nemia indicating a macrophage activation syndrome. A biopsy of nodules confirmed the diagnosis of sarcoidosis. The atypical EOS should be differentiated from histiocytosis.

UR - http://www.scopus.com/inward/record.url?scp=38549170011&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=38549170011&partnerID=8YFLogxK

U2 - 10.1002/pbc.21173

DO - 10.1002/pbc.21173

M3 - Article

C2 - 17366538

AN - SCOPUS:38549170011

VL - 50

SP - 723

EP - 726

JO - Pediatric Blood and Cancer

JF - Pediatric Blood and Cancer

SN - 1545-5009

IS - 3

ER -