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BMJ Case Rep 2019 May 5;12(5). Epub 2019 May 5.

Diagnostic and Interventional Radiology, University of Pisa, Pisa, Italy.

Eosinophilic granuloma (EG) is a rare bony disease deriving from abnormal proliferation of histiocytes, and is the most common form of presentation of Langerhans cell histiocytosis. EG predominantly affects the axial skeleton. However, when localised in the head and neck district, mandibular lesions account for the majority of cases. Read More

World Neurosurg 2019 05 13;125:301-311. Epub 2019 Feb 13.

Department of Orthodontics, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, India.

Objective: Spinal eosinophilic granulomas (EGs) are uncommon tumors, constituting <1% of all bone tumors. They are mostly seen in the pediatric age group, whereas adult onset is rare. The cervical spine is an infrequent location for EG. Read More

Cir Cir 2019 ;87(1):96-100

Delegación Baja California. Instituto Mexicano del Seguro Social, Baja California, México.

Background: Langerhans cell histiocytosis is a rare disease in the adult, predominates in the pediatric age and is more common in men than in women. The sites of greater affection are the long bones, followed by the bones of the skull. Patients usually have symptoms related to the site of affection, usually polyostotic lesions, monostotic lesions occur less frequently. Read More

BMC Oral Health 2018 06 8;18(1):106. Epub 2018 Jun 8.

Clinic of Cranio-Maxillofacial and Oral Surgery, University of Zurich, University Hospital Zurich, Plattenstrasse 11, CH-8032, Zurich, Switzerland.

Background: Bone necrosis of the jaw is a serious condition with a broad differential diagnosis of pathologies such as cutaneous histiocytosis, bone metastases or malignant tumours. In addition to the most common cause, medication related osteonecrosis of the jaw (MRONJ), one must consider a number of other causes, such as histiocytosis. Langerhans cell histiocytosis (LCH) is a histiocytic disorder with a large spectrum of clinical manifestations and with possible involvement of a variety of organs. Read More

Neurol India 2018 May-Jun;66(3):866-868

Department of Neurosurgery, Zhongshan City People's Hospital, Zhongshan, Guangdong, China.

Spec Care Dentist 2018 Mar 8;38(2):107-111. Epub 2018 Feb 8.

Department of Clinical and Experimental Medicine, University of Foggia, Foggia, Italy.

Langerhans cell histiocytosis (LCH) is a childhood pathology with a peak of incidence ranging from 1 to 4 years of age, though diagnosis is often made in adult age. LCH is clinically classified into three types: eosinophilic granuloma, Hand-Schuller-Christian disease and Abt-Letterer-Siwe disease. We report a case of Hand-Schüller-Christian disease with diabetes insipidus, skull and maxillary involvement in a 16-year-old boy referred to our observation for gradual increase in mobility of the teeth and subsequent gradual loss of the second premolars and the first molars of the upper jaw. Read More

Medicine (Baltimore) 2017 Dec;96(51):e9392

Department of Neurosurgery.

Rationale: Primary osteosarcomas of the skull and skull base are rare, comprising <2% of all skull tumors. Primary osteosarcomas of the skull are aggressive neoplasms composed of spindle cells producing osteoid which have poor outcome.

Patient Concerns: A 33-year-old woman was admitted to our hospital with a major complaint of a growing mass on her left frontal region of the skull for 10 months. Read More

J Dermatol 2018 Apr 15;45(4):491-495. Epub 2017 Dec 15.

Department of Dermatovenereology, West China Hospital, Sichuan University, Chengdu, China.

Histiocytoses are a group of heterogeneous diseases that encompass Langerhans cell histiocytosis and non-Langerhans cell histiocytosis. Cutaneous plane xanthoma is a non-Langerhans cell histiocytic disorder characterized by the presence of yellow-orange plaques on the face, neck, upper trunk and extremities. It can appear in association with several systemic diseases (including dyslipidemias, paraproteinemias, cardiovascular diseases and lymphoproliferative disorders), but is rarely connected with Langerhans cell histiocytoses. Read More

Arkh Patol 2017;79(4):33-39

Academician B.V. Petrovsky Russian Research Center of Surgery, Moscow, Russia.

The paper describes a case of eosinophilic granuloma of the parietal bone in a 32-year-old man. Histological examination revealed a large number of bean-shaped Langerhans cell histiocytes with lobed nuclei and nuclear grooves. The histiocytes alternated with the foci of obvious eosinophilic infiltration and with eosinophilic microabscesses. Read More

Int J Pediatr Otorhinolaryngol 2017 Aug 15;99:135-140. Epub 2017 Jun 15.

Department of Otolaryngology - Head and Neck Surgery, University of Texas Southwestern Medical Center, Dallas, TX, USA. Electronic address:

Objective: Describe the presentation, imaging characteristics, management, and outcomes of pediatric patients with Langerhans cell histiocytosis (LCH) of the temporal bone.

Methods: A retrospective chart review was performed between 2000 and 2014 at a single tertiary care children's hospital. Fourteen patients were identified with a diagnosis of LCH and involvement of the temporal bone. Read More

Medicine (Baltimore) 2017 Jun;96(25):e7197

aDepartment of Orthopedics, Union Hospital, Tongji Medical College of Huazhong University of Science and Technology, Wuhan, Hubei bDepartment of Ophthalmology, Shengjing Hospital of China Medical University, Shenyang, Liaoning cDepartment of Pathology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei, China.

Rationale: Solitary eosinophilic granuloma (EG), the most benign, common form of Langerhans cell histiocytosis, has a self-limiting process and is associated with a good prognosis. Immobilization is recommended as the first treatment strategy for solitary EG, although the treatment protocols are still controversial. Radiotherapy and surgery are secondary treatment choices. Read More

Cephalalgia 2018 04 10;38(4):794-797. Epub 2017 May 10.

1 Department of Neurology & Neurosurgery, Clinical Hospital of University of Chile, Santiago de Chile, Chile.

Background Nummular headache is a rare, recently described topographic headache defined by the circumscribed coin-shaped area of pain. It is classified as a primary headache. There is debate about whether it is due to a peripheral or central disturbance, and its relationship to migraine. Read More

Eur Spine J 2017 05 30;26(Suppl 1):225-228. Epub 2017 Mar 30.

Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.

Purpose: The eosinophilic granuloma is a unifocal or multifocal Langerhans cell histiocytosis characterized by an expanding proliferation of Langerhans cells in bones. Skeletal LCH is a rare condition, and vertebral regeneration in cases of vertebral body collapse is even rarer. We report the case of a girl with spontaneous complete healing. Read More

Childs Nerv Syst 2017 Apr 28;33(4):583-593. Epub 2017 Feb 28.

Department of Spine Surgery, Xiangya Hospital of Central South University, Xiangya Road 87, Changsha, Hunan Province, 410008, China.

Background: Eosinophilic granuloma (EG) of spine in pediatric patients presents kinds of clinical manifestation and a difficult management scenario. The choice of treatment, issues of surgical intervention versus conservative treatment, combination therapy or single treatment, all these factors, including neurological deficits, spinal stability, long-term complications, and continued skeletal growth, must be considered.

Methods: From 2008 to the 2015, 31 pediatric patients of spinal EG were retrospectively reviewed. Read More

Brain Pathol 2017 05;27(3):377-395

Department of Neurosurgery, University of Colorado Anschutz Medical Campus, Aurora, CO.

Xanthomatous lesions of the sellar region have traditionally been divided into two separate categories, xanthomatous hypophysitis (XH) and xanthogranuloma (XG) of the sellar region. The seminal article on XH, a condition typified by foamy histiocytes and lymphoplasmacytic infiltrates in the pituitary gland/sellar region, but usually little or no hemosiderin pigment, detailed three patients. However, most reports since that time have been single cases, making understanding of the entity difficult. Read More

J Orthop Traumatol 2017 Jun 21;18(2):83-90. Epub 2016 Oct 21.

Department of Orthopedics and Orthopedic Oncology, University of Padova, Padova, Italy.

This review summarizes current concepts in the diagnosis and management of the patients with eosinophilic granuloma. Given the benign biology, the clinical course, and the pediatric group of patients that this condition more commonly affects, a treatment approach that carries a lower risk of complications while ensuring a successful cure is desirable. Variable treatment options have been reported with satisfactory results and a recurrence rate of less than 20 %. Read More

J Neurosurg Spine 2017 Feb 7;26(2):252-256. Epub 2016 Oct 7.

Department of Neurosurgery, Washington University, Saint Louis, Missouri.

Spinal involvement occurs frequently in cases of eosinophilic granuloma (EG), but surgical treatment is limited primarily to those with spinal instability. Involvement of the cervical spine is rare, but primarily occurs in the vertebral bodies, and is normally amenable to anterior corpectomy and spinal reconstruction. The authors describe a 27-year-old man with pathologically proven EG who presented with complete destruction of the C-1 lateral mass requiring spinal stabilization. Read More

Eur Ann Otorhinolaryngol Head Neck Dis 2016 Dec 20;133(6):443-444. Epub 2016 Sep 20.

Service d'otologie et d'oto-neurotologie, hospices civils de Lyon, service d'otologie et d'oto-neurotologie, centre hospitalier Lyon Sud, 69495 Pierre-Bénite, France. Electronic address:

Pediatr Neurosurg 2017 6;52(1):41-45. Epub 2016 Sep 6.

Department of Neurosurgery, National Institute of Mental Health and Neurosciences, Bangalore, India.

Intracranial Langerhans cell histiocytosis commonly presents as skull lesions in children. An intratumoral cyst with fluid level and epidural hematoma occurring with eosinophilic granuloma is very rare. We report a 15-year-old boy who presented with a spontaneous epidural hematoma which was the result of a temporal eosinophilic granuloma. Read More

Biomed Res Int 2016 8;2016:2131859. Epub 2016 Aug 8.

Department of Orthopedics Surgery, Children's Hospital of SooChow University, Suzhou 215003, China.

Iliac bone destruction in children is uncommon and presents various imaging features. Correct diagnosis based on clinical and imaging features is difficult. This research aimed to retrospectively explore the clinical features, imaging, and histopathological diagnosis of children with iliac bone destruction. Read More

Neurosurg Focus 2016 Aug;41(2):E3

Department of Neurosurgery, Clinical Neurosciences Center and Huntsman Cancer Institute, University of Utah; and.

Spinal column tumors are rare in children and young adults, accounting for only 1% of all spine and spinal cord tumors combined. They often present diagnostic and therapeutic challenges. In this article, the authors review the current management of primary osseous tumors of the pediatric spinal column and highlight diagnosis, management, and surgical decision making. Read More

Head Face Med 2016 Jun 2;12(1):21. Epub 2016 Jun 2.

Observer of the Oncology and Maxillofacial Surgery Department, Medicine Faculty, Pontificia Universidad Católica de Chile, Santiago, Chile.

Background: Langerhans cell histiocytosis (LCH), previously known as Histiocytosis X, is an infrequent disease that congregates a wide spectrum of clinical presentations with variable systemic involvement. Unification of these diseases under only one category is based on the almost identical histopathologic features of the lesions, but the etiology and proper approach for each presentation remains controversial. The localized alternative of Langerhans cell histiocytosis (LLCH), known as Eosinophilic Granuloma (EG) of bone, is the predominant clinical presentation of LCH. Read More

World Neurosurg 2016 Jul 29;91:669.e15-9. Epub 2016 Mar 29.

Department of Neurosurgery, Erciyes University Faculty of Medicine, Kayseri, Turkey.

Background: The craniocervical junction is a complex anatomic location that contains the occipital bone, atlas, axis, and important complex ligamentous structures. The stability of this region is ensured only with the help of ligaments.

Case Description: A 6-year-old boy was admitted to our clinic for neck pain. Read More

J Craniofac Surg 2016 May;27(3):e255-7

Division of Maxillofacial Surgery, Head and Neck Department, San Giovanni Battista Hospital, University of Turin, Turin, Italy.

Eosinophilic granuloma is the most benign and common form of the Langerhans cell histiocytosis, a rare proliferative disease that can affect single or multiple organs. In the quite common head and neck manifestation these lesions can be confused in the beginning, with other bone diseases such as odontogenic cysts, periodontal disease, or malignancies. Treatment varies depending on the size, number, localization of the lesions, and patient's general conditions. Read More

Spine J 2016 Jan 16;16(1):e53-7. Epub 2015 Sep 16.

Department of Orthopaedic Surgery, Washington University, 660 S. Euclid Ave, Campus Box 8233, St. Louis, MO 63110, USA.

Background Context: Eosinophilic granulomas (EGs) of the sacrum have been reported in fewer than 10 patients. Treatment algorithms for these tumors remain poorly defined; there are no reports of treating solitary sacral EG with radiation therapy (RT).

Purpose: This study aimed to describe the presentation, treatment, and outcome of sacral EG in an adult patient with intractable pain and radiculopathy, treated in a novel fashion with RT. Read More

Acta Med Iran 2015 ;53(1):69-73

Department of Maxillofacial Radiology, School of Dentistry, Guilan University of Medical Sciences, Guilan, Iran.

This case report presents an eight-year-old girl having periauricular swelling and severe pain during mouth opening on the right-side temporomandibular joint (TMJ). CBCT showed extensive destruction of the base of the skull and the roof of the glenoid fossa on the right side. The findings based on CT and MRI images with and without contrast are discussed herein. Read More

BMJ Case Rep 2014 Apr 3;2014. Epub 2014 Apr 3.

Department of Pathology, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, UttarPradesh, India.

Eosinophilic granuloma (EG) is a rare histiocytic disorder resulting from clonal proliferation of Langerhans cells. It accounts for less than 1% of all osseous neoplasms and has a predilection for involving the axial skeleton. Although suspicion of the disease may arise from clinical features and radiographic demonstration of destructive bone lesions, it is still difficult to make a correct diagnosis without proper pathological evaluation. Read More

Spine J 2014 Nov 16;14(11):2701-9. Epub 2014 Mar 16.

Department of Orthopedics, Spine Tumor Center, ChangZheng Hospital, Second Military Medical University, 415 Fengyang Rd, Shanghai 200003, People's Republic of China. Electronic address:

Background Context: Eosinophilic granuloma (EG) involving the spine is uncommon in adults. Atlantoaxial instability (AAI) secondary to EG of the axis in adults is an extremely rare clinical condition that can give rise to severe neurologic morbidity or mortality if not treated appropriately. There have been no previous reports on the condition in adults. Read More

Gen Thorac Cardiovasc Surg 2014 Jul 11;62(7):434-40. Epub 2014 Mar 11.

Thoracic Surgery Department, 401 General Army Hospital, Athens, Greece,

Objective: A retrospective study of rib tumors was conducted to review their clinical, radiological, and pathological features, the difficulties in differentiating benign from malignant tumors, as well as the early and long-term results of surgical management.

Methods: All patients with rib lesions evaluated by the Thoracic Surgery Department from 1998 to 2012 were studied. The patient's age, sex, symptoms, radiologic evaluation, surgical procedure, pathologic diagnosis and follow-up were assessed. Read More

J Neurosurg Pediatr 2014 Mar 17;13(3):260-72. Epub 2014 Jan 17.

Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa.

Object: Atlantoaxial tumors account for a substantial proportion of primary bone tumors in children. Before resection, surgeons must consider the complex regional anatomy, the potential for neurological compromise, craniocervical instability, and the question of tumor resectability in a growing spine. Using current technology, the authors analyzed surgical cases in this light and present outcomes and treatment recommendations after long-term patient follow-up. Read More

Coll Antropol 2013 Jun;37(2):521-5

General Hospital Zabok, Department of Internal Medicine, Zabok, Croatia.

Our aim is to provide review of available studies on Langerhans cell histiocytosis (LCH) and discuss treatment for polyostotic monosystem form of disease based on our clinical experience. LCH is an enigmatic disease with insufficiently understood etiology, pathophysiology, and variety of clinical presentations ranging from solitary eosinophilic granuloma to severe multisystem disease. It is marked by formation of granuloma in practically any organ. Read More

Radiologe 2013 Oct;53(10):908-12

Diagnostische und Interventionelle Radiologie, Radiologische Klinik, Universitätsklinikum Heidelberg, Im Neuenheimer Feld 110, 69120, Heidelberg, Deutschland,

BMJ Case Rep 2013 May 22;2013. Epub 2013 May 22.

Department of Radiology, Sütçü Imam University Medical School, Kahramanmaraş, Turkey.

Eosinophilic granuloma (EG) is a well-recognised benign form of Langerhans-cell histiocytosis. The estimated incidence of EG is 3-4 per million of the population. More patients are children and adolescents between the ages of 1 and 15 years. Read More

J Neurosurg Pediatr 2013 Jul 10;12(1):1-5. Epub 2013 May 10.

Division of Hematology Oncology, Miami Children's Hospital Cancer Center and Herbert Wertheim College of Medicine, FL 33155, USA.

Object: Solitary eosinophilic granuloma (EG) of the calvaria is most commonly treated with surgical excision. The authors hypothesize that many solitary EGs will resolve without intervention, and observation may be a reasonable option. This study was undertaken to investigate that hypothesis. Read More

J Spinal Disord Tech 2013 Apr;26(2):E46-52

Department of Spine Surgery, The Second Xiangya Hospital of Central South University, Changsha, Hunan, PR China.

Study Design: This was a retrospective clinical study.

Objective: To evaluate the safety and accuracy of pedicle screw placement in very young children and to observe its influence on vertebral and spinal canal growth.

Summary Of Background Data: Although widely used, it is not known if pedicle screw fixation is safe and effective in very young children. Read More

Pediatr Int 2013 Feb;55(1):96-8

Departments of Pediatric Orthopedics Pediatric Radiology Pediatrics, Jichi Children's Medical Center Tochigi, Shimotsuke, Japan.

Eosinophilic granuloma, a type of Langerhans cell histiocytosis, exhibits a classic vertebral collapse, which is called vertebra plana (Calve's disease) and it manifests as a solitary bony lesion. Vertebra plana can cause severe pain in patients. Bisphosphonates (clodronate, pamidronate and zoledronic acid) have been recently used to treat osteolytic bone lesions of LCH. Read More

Coll Antropol 2012 Nov;36 Suppl 2:163-6

Dr. Josip Bencević General Hospital, Department of Otorhinolaryngology Head and Neck Surgery, Slavonski Brod, Croatia.

Eosinophilic granuloma is a rare, benign, lesion characterized by uncontrolled proliferation of Langerhan's cells. It is commonly found in a bone and has been described in almost every bone in the body. It usually affects children and is seldom found in adults. Read More

Orthopedics 2012 Dec;35(12):e1818-21

Department of Orthopedic Surgery, Fukui Prefectural Hospital, 2-8-1, Yotsui, Fukui 9108526, Japan.

Eosinophilic granuloma of the spine is a common benign disease in children and adolescents that rarely affects adults.This article describes the case of a 32-year-old woman with a solitary eosinophilic granuloma presenting as a local lytic lesion at the L4 vertebral body. She presented with a 2-month history of low back pain without neurological deficits. Read More

Magn Reson Imaging 2013 Apr 25;31(3):418-31. Epub 2012 Oct 25.

Department of Radiology, Yale University-Bridgeport hospital, Bridgeport, CT 06610, USA.

Magnetic Resonance Imaging (MRI) is the preferred modality of choice to image epiphyseal lesions. It provides excellent soft tissue resolution and extent of disease. A wide spectrum of tumor and tumor like lesions can involve the epiphysis. Read More

Brain Dev 2013 Apr 12;35(4):372-5. Epub 2012 Jul 12.

Department of Pediatrics, Shizuoka Municipal Hospital, Shizuoka, Japan.

Eosinophilic granuloma (EG) is a benign, self-limiting disorder that usually involves a single bone. However, there is a growing evidence that the clinical picture of EG is protean. We report two cases with EG that showed rare presentations. Read More

J Pediatr Orthop 2012 Jun;32(4):416-22

Department of Orthopedics, Istituto Ortopedico Rizzoli, University of Bologna, Bologna, Italy.

Background: Previous case reports and small series have reported on the treatment of eosinophilic granuloma of bone. We present our long experience in a large group of children and teenagers with symptomatic eosinophilic granuloma of the appendicular skeleton to evaluate clinical and imaging outcome after methylprednisolone injection.

Methods: Sixty-six patients with symptomatic solitary eosinophilic granuloma of the appendicular skeleton treated by incisional or percutaneous biopsy and methylprednisolone injection were retrospectively studied. Read More

J Neurosurg Sci 2012 Mar;56(1):67-72

Department of Neurosurgery, University of Siena, Siena, Italy.

Eosinophilic granuloma (EG), Letterer-Siwe disease and Hand-Schuller-Christian disease are collectively called Langherans-cell histiocytosis (LCH). While the latter two are systemic diseases, the former is a localized form of histiocytosis. Solitary EG of the skull are rare lesions characterized by a natural history not well defined yet. Read More

Am J Hematol 2012 Jun 27;87(6):626-7. Epub 2011 Dec 27.

Department of Pathology, Duke University Medical Center, Durham, North Carolina, USA.

Eur Spine J 2012 Jun 30;21 Suppl 4:S441-4. Epub 2011 Oct 30.

Department of Oncologic and Degenerative Spine Surgery, Rizzoli Institute, Via Pupilli 1, 40136, Bologna, Italy.

Introduction: Eosinophilic granuloma (EG) is a benign bone tumor that rarely occurs in adults. It is usually found occurring in flat and long bones, but spine is often affected too. EG is of unknown aetiology, and the course of the disease is unpredictable. Read More

Fetal Pediatr Pathol 2011 21;30(6):380-6. Epub 2011 Oct 21.

Department of Orthopaedic Surgery, The First Affiliated Hospital of Sun Yat-Sen University, Guang Zhou, China.

A 15-year-old boy presented with a right shoulder pain and the inability to use his right upper limb after carrying a heavy object. Physical examination and local imaging revealed a benign tumor or tumor-like lesion in the upper ends of both humeri and a pathologic fracture in the right humerus. An initial biopsy was reported as fibroblastic osteosarcoma. Read More

Dtsch Med Wochenschr 2011 Oct 28;136(40):2040-2. Epub 2011 Sep 28.

Diagnostische und interventionelle Radiologie, HELIOS-Klinikum Wuppertal, Universitätsklinik Witten/Herdecke.

History And Clinical Findings: A 69-year-old woman was admitted for evaluation of a left occipital subcutaneous tumour which had grown during the preceding eight weeks from 2 × 2 cm to 4 × 4 cm.

Investigations: Sonography revealed a pressure-sensitive subcutaneous mass with osteolytic destruction in the occipital bone. Cranial magnetic resonance imaging confirmed the osteolytic lesion. Read More

Acta Orthop Traumatol Turc 2011 ;45(1):66-9

Department of Orthopedics and Traumatology, Bezmialem Vakif University, ‹stanbul, Turkey.

Osteoid osteoma, a common bone lesion of benign nature, is more rarely seen in feet. It most commonly involves the talus yet rarely the cuboid. The atypical symptoms of foot involvement may delay the diagnosis. Read More

Kaohsiung J Med Sci 2011 Feb 12;27(2):76-9. Epub 2011 Feb 12.

Division of Neurosurgery, Department of Surgery, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan.

Eosinophilic granuloma (EG) refers to the most common and benign form of the disorder known as Langerhans' cell histiocytosis. The disease is typically found in children and adolescents and rarely affects adults. We present a case of EG in the occipital bone in a 36-year-old man, who visited our hospital with the chief complaint of left occipital palpable tumor mass with local tenderness and pain for one month. Read More

An Pediatr (Barc) 2011 Mar;74(3):212-4

J Orthop Sports Phys Ther 2011 Feb 31;41(2):119. Epub 2011 Jan 31.

Naval Special Warfare Group FOUR, Virginia Beach, VA, USA.