The Journal of Bone & Joint Surgery

1997

DOI: 10.2106/00004623-199706000-00017

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Sclerotic Lesion of the Tibia without Involvement of Lymph Nodes. Report of an Unusual Case of Rosai-Dorfman Disease*

Francis Patterson

¹

,

Michelle Nayahamui Rooney

²

,

Timothy A. Damron

³

et al.

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Cited by 25 publications

(24 citation statements)

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“…Lymphoma of bone often involves the diaphysis and presents with localized bone pain, but it typically appears radiographically as a more diffuse, permeative lytic process, often with an associated soft tissue mass [14]. RosaiDorfman disease typically presents with bilateral cervical lymphadenopathy, but in 2% of cases can present as isolated extranodal bone disease [16,19]. In cases with bone involvement, osteolytic lesions predominate [16,19].…”

Section: Discussion and Treatmentmentioning

confidence: 99%

“…RosaiDorfman disease typically presents with bilateral cervical lymphadenopathy, but in 2% of cases can present as isolated extranodal bone disease [16,19]. In cases with bone involvement, osteolytic lesions predominate [16,19]. Histologically, Rosai-Dorfman disease is characterized by large histiocytes showing emperipolesis (lymphocytophagocytosis).…”

Section: Discussion and Treatmentmentioning

confidence: 99%

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Orthopaedic Case of the Month: Recurrent Thigh Pain in a 44-Year-Old Man

¹

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²

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³

2012

Clinical Orthopaedics &Amp; Related Research

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A 44-year-old man presented with progressively worsening left thigh pain of 8 years' duration. The patient's medical history was significant for a diagnosis of osteoid osteoma of the left femur treated elsewhere 24 years previously. At that time, the patient underwent open incisional biopsy ( Fig. 1), curettage, and autogenous iliac crest bone grafting without stabilization. The patient had no complications after surgery and was in good health until 8 years before this visit when he began to experience intermittent left thigh pain which gradually increased in frequency and severity. By the time the patient presented to us, he described a dull pain present most of the time punctuated by intermittent sharp pain. The patient could not identify any particular palliative or provocative factors for the pain. Acetaminophen provided minimal relief. He had experienced some decrease in his energy level but denied any fevers, chills, night sweats, skin changes, abdominal pain, diarrhea, unintentional weight loss, or decrease in appetite.On physical examination, the patient appeared healthy and had a normal gait. He had full active and passive ROM of the left hip, knee, and ankle without limitation. Motor testing and sensation in the left lower extremity were normal. Distal pulses were present and normal in the left lower extremity. There was exquisite tenderness to palpation over a several-centimeter diameter region of the anterior and lateral left mid-thigh, but there was no palpable mass in that extremity or any skin lesions at this site or elsewhere. The patient had increased thigh pain with provocative testing, including resisted hip flexion, extremes of hip rotation, and with full passive hip flexion.One month before presentation to our institution the patient had sought care from his primary care physician. The physician ordered radiographs (Fig. 2). A CT scan (Fig. 3) and bone scan (Fig. 4) were obtained subsequently and the patient was sent to us for additional orthopaedic care.Based on the history, physical examination, and imaging studies, what is the differential diagnosis at this point? Imaging InterpretationThe radiographs (Fig. 2) and CT images (Fig. 3) of the left femur showed a geographic 3-cm diaphyseal radiolucency

“…Lymphoma of bone often involves the diaphysis and presents with localized bone pain, but it typically appears radiographically as a more diffuse, permeative lytic process, often with an associated soft tissue mass [14]. RosaiDorfman disease typically presents with bilateral cervical lymphadenopathy, but in 2% of cases can present as isolated extranodal bone disease [16,19]. In cases with bone involvement, osteolytic lesions predominate [16,19].…”

Section: Discussion and Treatmentmentioning

confidence: 99%

“…RosaiDorfman disease typically presents with bilateral cervical lymphadenopathy, but in 2% of cases can present as isolated extranodal bone disease [16,19]. In cases with bone involvement, osteolytic lesions predominate [16,19]. Histologically, Rosai-Dorfman disease is characterized by large histiocytes showing emperipolesis (lymphocytophagocytosis).…”

Section: Discussion and Treatmentmentioning

confidence: 99%

Orthopaedic Case of the Month: Recurrent Thigh Pain in a 44-Year-Old Man

¹

,

²

,

³

2012

Clinical Orthopaedics &Amp; Related Research

Self Cite

View full text Add to dashboard Cite

A 44-year-old man presented with progressively worsening left thigh pain of 8 years' duration. The patient's medical history was significant for a diagnosis of osteoid osteoma of the left femur treated elsewhere 24 years previously. At that time, the patient underwent open incisional biopsy ( Fig. 1), curettage, and autogenous iliac crest bone grafting without stabilization. The patient had no complications after surgery and was in good health until 8 years before this visit when he began to experience intermittent left thigh pain which gradually increased in frequency and severity. By the time the patient presented to us, he described a dull pain present most of the time punctuated by intermittent sharp pain. The patient could not identify any particular palliative or provocative factors for the pain. Acetaminophen provided minimal relief. He had experienced some decrease in his energy level but denied any fevers, chills, night sweats, skin changes, abdominal pain, diarrhea, unintentional weight loss, or decrease in appetite.On physical examination, the patient appeared healthy and had a normal gait. He had full active and passive ROM of the left hip, knee, and ankle without limitation. Motor testing and sensation in the left lower extremity were normal. Distal pulses were present and normal in the left lower extremity. There was exquisite tenderness to palpation over a several-centimeter diameter region of the anterior and lateral left mid-thigh, but there was no palpable mass in that extremity or any skin lesions at this site or elsewhere. The patient had increased thigh pain with provocative testing, including resisted hip flexion, extremes of hip rotation, and with full passive hip flexion.One month before presentation to our institution the patient had sought care from his primary care physician. The physician ordered radiographs (Fig. 2). A CT scan (Fig. 3) and bone scan (Fig. 4) were obtained subsequently and the patient was sent to us for additional orthopaedic care.Based on the history, physical examination, and imaging studies, what is the differential diagnosis at this point? Imaging InterpretationThe radiographs (Fig. 2) and CT images (Fig. 3) of the left femur showed a geographic 3-cm diaphyseal radiolucency

“…A mixed population of T and B lymphocytes and immunostaining excluded a chronic lymphocytic leukemia/ small lymphocytic lymphoma. Immunostaining ruled out Langerhans cell histiocytosis (CD1A positive) and Erdheim-Chester disease (CD1a and S-100 negative) [1,2,6,7,9]. Infection was ruled out by normal serum testing, MRI showing no reactive edema or discrete fluid collection, and negative cultures.…”

Section: Discussion and Treatmentmentioning

confidence: 99%

“…Approximately 8% of extranodal cases involve bone with less than 1% having isolated osseous involvement [3]. Bone lesions may be seen anywhere, but the skull, femur, and tibia are the most commonly involved [2,3,9,10]. The mean age of patients at onset is between 20 to 28 years [2,3].…”

Section: Discussion and Treatmentmentioning

confidence: 99%

“…Patients typically present with unimproved pain after antecedent injury, but RDD may be found incidentally [1,6,7,9]. Palpable pain and notable swelling are often apparent [2].…”

Section: Discussion and Treatmentmentioning

confidence: 99%

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Orthopaedic Case of the Month: A 50-year-old Woman with Persistent Knee Pain

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et al. 2011

Clinical Orthopaedics &Amp; Related Research

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Faisalabad histiocytosis mimics Rosai‐Dorfman disease: Brothers with lymphadenopathy, intrauterine fractures, short stature, and sensorineural deafness

¹

,

²

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³

et al. 2005

Pediatric Blood & Cancer

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Rosai-Dorfman disease (RDD) is a rare, sporadic histiocytic disorder characterized by painless but protracted lymphadenopathy. Its etiology remains unclear. The observation of congenital disease and reports of familial cases with seven pairs of siblings including three sets of identical twins suggests a genetic predisposition in some patients with this condition. We now report two brothers of consanguineous Palestinian parents, whose lymphadenopathy, lymph node histology, and polyclonal hypergammaglobulinemia indicated RDD. The presence of intrauterine fractures, short stature, and sensorineural hearing impairment suggested a rare familial form of the disorder. Moynihan et al. recently described a Pakistani family with a familial histiocytic disorder highly reminiscent of the brothers reported here, whose lymph node morphology was apparently consistent with RDD as well. The presence of sensorineural deafness, short stature, and joint contractures, however, suggested a separate, rare autosomal recessive syndrome referred to as Faisalabad histiocytosis, after the family's place of origin. We believe that the brothers described here represent a second family with Faisalabad histiocytosis, which mimics RDD histologically.

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