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Bone pathologic fracture revealing an unusual association: coexistence of Langerhans cell histiocytosis with Rosai-Dorfman disease

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ABSTRACT

Background: The coexistence of Rosai-Dorfman disease (RDD) with Langerhans cell histiocytosis (LCH) is very rare, as to date only 17 cases have been reported in the english literature. The pathophysiology of this uncommon co-occurrence still remains enigmatic and a subject of various speculations.

Case presentation: We report a case of a 30-year-old female patient who presented with a pathologic fracture of the left proximal femur. Her medical history was unremarkable, there were no fever, skin lesions, lymphadenopathy or other organomegaly at physical examination. X-ray radiograph of the fractured femur showed an isolated and ill-defined osteolytic lesion. The histopathological analysis of biopsies from this lesion were consistent with a combined RDD-LCH of the bone.

Conclusion: Combined RDD-LCH is a very rare phenomenon, whose pathophysiology still remains unclear and a subject of various speculations.

No MeSH data available.


Related in: MedlinePlus

X-rays of the hip showing an osteolytic tumor at the site of the fracture (left side)
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Fig1: X-rays of the hip showing an osteolytic tumor at the site of the fracture (left side)

Mentions: A 30-year-old woman was admitted at emergency department for a pathologic fracture of the left femur bone, after a mild trauma. The patient’s medical history, as well as her family history were unremarkable. The physical examination showed a painful and swollen left hip with inability to move the lower limb because of the pain. Apart from these trauma-related signs, there were no fever, skin lesions, lymphadenopathy or other organomegaly. An X-ray radiograph has been performed and revealed at the traumatic site, a relatively well-limited trochanteric osteolityc lesions at the fracture’s site (Fig. 1). No other lesions have been discovered. The biological check-up was within normal limits. Biopsies have been performed for histopathological evaluation.Fig. 1


Bone pathologic fracture revealing an unusual association: coexistence of Langerhans cell histiocytosis with Rosai-Dorfman disease
X-rays of the hip showing an osteolytic tumor at the site of the fracture (left side)
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC5383940&req=5

Fig1: X-rays of the hip showing an osteolytic tumor at the site of the fracture (left side)
Mentions: A 30-year-old woman was admitted at emergency department for a pathologic fracture of the left femur bone, after a mild trauma. The patient’s medical history, as well as her family history were unremarkable. The physical examination showed a painful and swollen left hip with inability to move the lower limb because of the pain. Apart from these trauma-related signs, there were no fever, skin lesions, lymphadenopathy or other organomegaly. An X-ray radiograph has been performed and revealed at the traumatic site, a relatively well-limited trochanteric osteolityc lesions at the fracture’s site (Fig. 1). No other lesions have been discovered. The biological check-up was within normal limits. Biopsies have been performed for histopathological evaluation.Fig. 1

View Article: PubMed Central - PubMed

ABSTRACT

Background: The coexistence of Rosai-Dorfman disease (RDD) with Langerhans cell histiocytosis (LCH) is very rare, as to date only 17 cases have been reported in the english literature. The pathophysiology of this uncommon co-occurrence still remains enigmatic and a subject of various speculations.

Case presentation: We report a case of a 30-year-old female patient who presented with a pathologic fracture of the left proximal femur. Her medical history was unremarkable, there were no fever, skin lesions, lymphadenopathy or other organomegaly at physical examination. X-ray radiograph of the fractured femur showed an isolated and ill-defined osteolytic lesion. The histopathological analysis of biopsies from this lesion were consistent with a combined RDD-LCH of the bone.

Conclusion: Combined RDD-LCH is a very rare phenomenon, whose pathophysiology still remains unclear and a subject of various speculations.

No MeSH data available.


Related in: MedlinePlus