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Pediatr Blood Cancer 2021 Jun 9:e29140. Epub 2021 Jun 9.

French Reference Center for Langerhans Cell Histiocytosis, Trousseau Hospital, Paris, France.

Background: The somatic BRAF mutation occurs in 38-64% of pediatric cases of Langerhans cell histiocytosis (LCH). Vemurafenib (VMF), a BRAF inhibitor, was approved for refractory BRAF mutated LCH. In adults, VMF causes frequent cutaneous adverse events (CAE) including skin tumors (squamous cell carcinomas, melanomas), but little is known in children. Read More

JAMA Dermatol 2021 May 5. Epub 2021 May 5.

Division of Rheumatology, Department of Medicine, Inflammation and Immunity Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts.

Joint Bone Spine 2021 Apr 20;88(5):105192. Epub 2021 Apr 20.

Department of Dermatology, APHP, Saint-Louis Hospital, Université de Paris, 1, avenue Claude Vellefaux, 75010 Paris, France. Electronic address:

Dermatol Online J 2021 Mar 15;27(3). Epub 2021 Mar 15.

Department of Dermatology, HCA Healthcare/USF Morsani College of Medicine Largo, FL.

Multicentric reticulohistiocytosis (MRH) is a rare type of non-Langerhans cell histiocytosis characterized by coral-toned papules with predilection for dorsal surfaces in addition to severe arthropathy. It sometimes proves difficult to differentiate these joint and skin findings clinically from certain rheumatologic diseases, primarily dermatomyositis. Herein, we present an 82-year-old woman who presented with the clinical findings described above and was subsequently diagnosed with MRH after biopsy and review of relevant clinical history. Read More

Skeletal Radiol 2021 Mar 31. Epub 2021 Mar 31.

Department of Radiology, Royal National Orthopaedic Hospital, Brockley Hill, Stanmore, Middlesex, HA7 4LP, UK.

The non-Langerhans cell histiocytoses (N-LCH) represent a group of rare diseases with different clinical presentations and imaging features to classical LCH. While there is a long list of entities, only few present with musculoskeletal soft tissue and osseous manifestations alongside the more commonly reported systemic findings. Erdheim-Chester disease (ECD) is typically seen in adults as bilateral and symmetrical long bone osteosclerosis. Read More

JAAD Case Rep 2021 Mar 25;9:18-20. Epub 2020 Dec 25.

Dermatology and Venereology Department, Military Medical Academy, Sofia, Bulgaria.

BMJ Case Rep 2021 Jan 25;14(1). Epub 2021 Jan 25.

Pathology, Kangwon National University Hospital, Chuncheon, Kangwondo, Korea (the Republic of).

We report an extremely rare case of adult Langerhans cell histiocytosis (LCH) in a patient with papillary thyroid carcinoma (PTC) and Castleman disease (CD). A 46-year-old man visited our hospital with anaemia; systemic imaging showed an abdominal and a left thyroid mass. Biopsy confirmed CD for the abdominal mass and PTC for the thyroid mass. Read More

J Clin Rheumatol 2020 Dec 15;Publish Ahead of Print. Epub 2020 Dec 15.

From the Department of Dermatology, Fukushima Medical University, Fukushima, Japan.

Abstract: Multicentric reticulohistiocytosis (MRH) is a rare multisystem disorder, primarily affecting the skin and joints. As severe joint damage is a possible symptom, early diagnosis and therapeutic intervention are required. Cutaneous lesions present with characteristic features such as discrete reddish nodules, especially on acral locations. Read More

Clin Nucl Med 2021 May;46(5):e253-e255

From the Service de Médecine Nucléaire.

Abstract: We report the case of a 60-year-old woman who underwent 18F-FDG PET/CT to evaluate a metastatic breast carcinoma. Follow-up 18F-FDG PET/CT showed progressive disease with 18F-FDG increased in primary tumor, axillary lymph nodes, and pleural and bone diffuse metastases but also a concomitant uptake in multiples joints. The anatomopathological analysis from skin biopsy revealed a multicentric reticulohistiocytosis, considered paraneoplastic in the context. Read More

Indian Dermatol Online J 2020 Sep-Oct;11(5):849-851. Epub 2020 Sep 19.

Department of Dermatology, Maulana Azad Medical College, Bahadur Shah Zafar Marg, Delhi, India.

Medicina (Kaunas) 2020 Sep 8;56(9). Epub 2020 Sep 8.

Department of Internal Medicine, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, 16 Universității Street, 700115 Iași, Romania.

Multicentric reticulohistiocytosis (MRH) is a rare cause of destructive inflammatory arthritis involving both small, as well as larger joints. We report the case of a 40-year-old Caucasian female with a family history of neoplasia who was referred to our service witha two-month history of inflammatory joint pain. On examination, the patient had inflammatory arthritis, mainly involving the peripheral joints, sacroiliac joint pain, and numerous papulonodular mucocutaneous lesions, including periungual "coral beads". Read More

Clin Case Rep 2020 Aug 22;8(8):1560-1563. Epub 2020 Jun 22.

Department of Dermatology UCSF-Fresno Fresno CA USA.

Although MRH can mimic rheumatoid arthritis, its ability to rapidly progress to arthritis mutilans and association with malignancy in up to 25% of patients warrant prompt recognition and treatment along with age-appropriate malignancy work-up. Read More

Clin Exp Dermatol 2021 Apr 10;46(3):554-556. Epub 2020 Oct 10.

Academic Rheumatology, Clinical Sciences Building, City Hospital Nottingham, Nottingham, UK.

Eur J Case Rep Intern Med 2020 25;7(8):001568. Epub 2020 May 25.

FTC, Faculdade de Tecnologia e Ciências, Salvador, Bahia, Brazil.

Objectives: We describe a case of systemic sclerosis (SS) with acro-osteolysis associated with cutaneous mucinosis, usually characterized by mucin deposition in the skin. The main differential diagnosis was multicentric reticulohistiocytosis due to the presentation of papulonodular skin lesions.

Materials And Methods: A physical examination, imaging studies and laboratory tests were performed. Read More

Intern Med 2020 09 9;59(18):2337-2338. Epub 2020 Jun 9.

Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Japan.

Blood Rev 2021 01 15;45:100707. Epub 2020 May 15.

Division of Hematology, University of British Columbia, Canada.

A subset of patients with severe COVID-19 develop profound inflammation and multi-organ dysfunction consistent with a "Cytokine Storm Syndrome" (CSS). In this review we compare the clinical features, diagnosis, and pathogenesis of COVID-CSS with other hematological CSS, namely secondary hemophagocytic lymphohistiocytosis (sHLH), idiopathic multicentric Castleman disease (iMCD), and CAR-T cell therapy associated Cytokine Release Syndrome (CRS). Novel therapeutics targeting cytokines or inhibiting cell signaling pathways have now become the mainstay of treatment in these CSS. Read More

Med Clin (Barc) 2021 03 12;156(6):310-311. Epub 2020 Mar 12.

Servicio de Dermatología, Hospital Royo Villanova, Zaragoza, España.

Oncogene 2020 04 12;39(15):3218-3225. Epub 2020 Feb 12.

Human Oncology and Pathogenesis Program, Memorial Sloan Kettering Cancer Center, New York, NY, 10065, USA.

TAFRO syndrome, a clinical subtype of idiopathic multicentric Castleman disease (iMCD), consists of a constellation of symptoms/signs including thrombocytopenia, anasarca, fever, reticulin fibrosis/renal dysfunction, and organomegaly. The etiology of iMCD-TAFRO and the basis for cytokine hypersecretion commonly seen in iMCD-TAFRO patients has not been elucidated. Here, we identified a somatic MEK2 mutation and a germline RUNX1 mutation in two patients with iMCD-TAFRO, respectively. Read More

J Eur Acad Dermatol Venereol 2020 Aug 19;34(8):1684-1694. Epub 2020 May 19.

Unit of Dermatology, IRCCS Ca' Granda - Maggiore Policlinico Hospital Foundation, University of Milan, Milan, Italy.

Reticulohistiocytoses (RH) are rare and clinically heterogeneous histiocytic disorders of dermatological interest. Three clinical entities with superimposable histopathological features are currently considered, namely solitary reticulohistiocytoma, diffuse/generalized reticulohistiocytosis and multicentric reticulohistiocytosis. Although in the last decade, RH studies have only minimally progressed, histiocytosis research has advanced considerably: the prognostic and therapeutic importance of the clinical subclassification of histiocytosis patients as well as of the detection of genetic alterations in the genes of the ERK pathway has been highlighted. Read More

Radiographics 2020 Jan-Feb;40(1):223-240

From the Division of Nuclear Medicine, Department of Radiology (M.H., R.M.), and Division of Rheumatic Diseases (H.K., H.Y.), National Center for Global Health and Medicine, 1-21-1 Toyama, Shinjuku-ku, Tokyo 162-8655, Japan.

Rheumatic diseases are various painful conditions that affect joints, bones, cartilage, tendons, ligaments, and muscles. Arthritis is a typical condition of rheumatic disease. Although rheumatoid arthritis is a representative rheumatic disease, various diseases other than rheumatoid arthritis can also affect joints, and differential diagnosis of rheumatic diseases is often difficult owing to the similar clinical manifestations. Read More

Postepy Dermatol Alergol 2019 Oct 12;36(5):646-647. Epub 2019 Nov 12.

Department of Rheumatology and Internal Diseases, Poznan University of Medical Sciences, Poznan, Poland.

Int J Dermatol 2020 Jun 28;59(6):e203-e204. Epub 2019 Nov 28.

Division of Rheumatology, Allergy & Clinical Immunology, School of Medicine, University of California, Davis, CA, USA.

Rheumatology (Oxford) 2020 08;59(8):1898-1905

Division of Rheumatology, Department of Internal Medicine, Mayo Clinic, Rochester, MN.

Objectives: Multicentric reticulohistiocytosis (MRH), a rare histiocytic disease that can mimic other rheumatic conditions, may be associated with cancer and other autoimmune disorders. To better understand the disorder and its other associations, we aimed to evaluate clinical correlates and outcomes of all patients with MRH seen at Mayo Clinic, Rochester between 1980 and 2017.

Methods: A retrospective medical record review was conducted to identify all patients with MRH between 1 January 1980 and 30 April 2017. Read More

Cureus 2019 Aug 24;11(8):e5476. Epub 2019 Aug 24.

Pathology, All India Institute of Medical Sciences, Bhubaneswar, IND.

Multicentric reticulohistiocytosis (MRH) is a rare multisystem macrophage disorder of unknown etiology characterized by papulonodular skin and mucosal lesions, rapidly progressive erosive symmetric polyarthritis, and inflammation of internal organs. Most often, it is misdiagnosed as rheumatoid arthritis (RA). Here, we report the case of a 60-year-old woman found to have features of both MRH and RA with positive rheumatoid factor and high titer of anti-cyclic citrullinated peptide antibody in serum. Read More

JAAD Case Rep 2019 Aug 5;5(8):720-722. Epub 2019 Aug 5.

Department of Dermatology, University of California Irvine, Irvine, California.

J Hand Surg Am 2020 May 15;45(5):457.e1-457.e5. Epub 2019 Jul 15.

Department of Orthopaedic Surgery, McLaren Regional Medical Center, Flint, MI.

Multicentric reticulohistiocytosis is a rare non-Langerhans cell histiocytic rheumatologic disorder characterized by multiple skin lesions, painful hand nodules, and destructive polyarthritis that results in arthritis mutilans in 20% to 30% of patients. In the current literature, there have been approximately 300 cases reported and the pathogenesis remains unclear. Currently, there is no diagnostic serologic test and no consensus on management. Read More

Haematologica 2020 31;105(2):e61-e64. Epub 2020 Jan 31.

Department of Rehabilitation, Nagoya University Hospital, Nagoya, Japan

J Dtsch Dermatol Ges 2019 Jul 30;17(7):746-748. Epub 2019 May 30.

Department of Dermatology, Venereology and Allergology, University Medical Center Ruprecht-Karls-University Heidelberg, Germany.

Acta Derm Venereol 2019 Jul;99(9):844-845

Department of Dermatology, University of Padova, IT-35128 Padova, Italy.

Pediatr Blood Cancer 2019 07 15;66(7):e27759. Epub 2019 Apr 15.

Beijing Key Laboratory of Pediatric Hematology Oncology, National Key Discipline of Pediatrics (Capital Medical University), Key Laboratory of Major Diseases in Children, Ministry of Education; Hematology Oncology Center, Beijing Children's Hospital, Capital Medical University, National Center for Children's Health, Beijing, China.