Eosinophilic colitis accompanied by Tolosa–Hunt syndrome: report of a case

Kosugi, Shin‐ichi; Date, Kazutoshi; Minagawa, Masahiro; Ishikawa, Hiroyuki; Hatakeyama, Katsuyoshi; Endo, Kenji; Kimura, Yoshihiko

doi:10.1007/s00535-003-1130-y

Cited by 9 publications

(7 citation statements)

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“…ChurgStrauss syndrome). High serum IgE level (1300 U/ml) and marked submucosal infiltration with eosinophils arise in the TolosaHunt syndrome, a rare neurological disorder characterized by headaches, ophthalmoplegia, and cranial nerve palsies [Kosugi et al 2003]. Primary EC remains therefore a diagnosis of exclusion (Table 3).…”

Section: Eosinophilic Esophagitismentioning

confidence: 99%

Eosinophilic colitis: epidemiology, clinical features, and current management

Alfadda

Storr

Shaffer

2010

Therap Adv Gastroenterol

117

137

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Primary eosinophilic gastrointestinal disorders (EGIDs) represent a spectrum of inflammatory gastrointestinal disorders in which eosinophils infiltrate the gut in the absence of known causes for such tissue eosinophilia. EGIDs can be subgrouped as eosinophilic esophagitis (EE), eosinophilic gastroenteritis (EG), and eosinophilic colitis (EC). The least frequent manifestation of EGIDs is EC. EC is a heterogeneous entity with a bimodal age distribution, presenting with either an acute self-limited bloody diarrhea in otherwise healthy infants or as a more chronic relapsing colitis in young adults. The pathophysiology of primary EC appears related to altered hypersensitivity, principally as a food allergy in infants and T lymphocytemediated (i.e. non-IgE associated) in young adults. In adults, symptoms include diarrhea, abdominal pain, and weight loss. Endoscopic changes are generally modest, featuring edema and patchy granularity. Although standardized criteria are not yet established, the diagnosis of EC depends on histopathology that identifies an excess of eosinophils. Therapeutic approaches are based on case reports and small case series, as prospective randomized controlled trials are lacking. Eosinophilic colitis in infants is a rather benign, frequently food-related entity and dietary elimination of the aggressor often resolves the disorder within days. Adolescent or older patients require more aggressive medical management including: glucocorticoids, antihistamines, leukotriene receptors antagonists as well as novel approaches employing biologics that target interleukin-5 (IL-5) and IgE. This review article summarizes the current knowledge of EC, its epidemiology, clinical manifestations, diagnosis, and treatment.

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Section: Eosinophilic Esophagitismentioning

confidence: 99%

Eosinophilic colitis: epidemiology, clinical features, and current management

Alfadda

Storr

Shaffer

2010

Therap Adv Gastroenterol

117

137

View full text Add to dashboard Cite

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“…Drug-induced EC has also been described in response to several medications, including clozapine, carbamazepine, rifampicin, nonsteroidal antiinflammatory agents, tacrolimus, and gold [19–25]. Other conditions associated with EC include systemic mastocytosis, malignancy, autoimmune connective tissue disease including scleroderma, dermatomyositis and polymyositis, [9, 26, 27] allogeneic bone marrow transplantation [28], and the rare Tolosa-Hunt syndrome that features inflammatory ophthalmoparesis [29]. Idiopathic hypereosinophilic syndrome may also affect the colon, but this rare condition presents with sustained and marked peripheral eosinophilia with end-organ damage that extends beyond the gastrointestinal tract (e.g., heart and skin) [30].…”

Section: Diagnosismentioning

confidence: 99%

Eosinophilic Colitis: University of Minnesota Experience and Literature Review

Gaertner

MacDonald

Kwaan

et al. 2011

Gastroenterology Research and Practice

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Eosinophilic colitis is a rare form of primary eosinophilic gastrointestinal disease that is poorly understood. Neonates and young adults are more frequently affected. Clinical presentation is highly variable depending on the depth of inflammatory response (mucosal, transmural, or serosal). The pathophysiology of eosinophilic colitis is unclear but is suspected to be related to a hypersensitivity reaction given its correlation with other atopic disorders and clinical response to corticosteroid therapy. Diagnosis is that of exclusion and differential diagnoses are many because colonic tissue eosinophilia may occur with other colitides (parasitic, drug-induced, inflammatory bowel disease, and various connective tissue disorders). Similar to other eosinophilic gastrointestinal disorders, steroid-based therapy and diet modification achieve very good and durable responses. In this paper, we present our experience with this rare pathology. Five patients (3 pediatric and 2 adults) presented with diarrhea and hematochezia. Mean age at presentation was 26 years. Mean duration of symptoms before pathologic diagnosis was 8 months. Mean eosinophil count per patient was 31 per high-power field. The pediatric patients responded very well to dietary modifications, with no recurrences. The adult patients were treated with steroids and did not respond. Overall mean followup was 22 (range, 2–48) months.

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“…Interestingly, the eosinophilic infiltrates in that patient were transmural and were at their maximum in the transverse colon with more distal diminution toward the rectosigmoid, so the reason for localization of polyps exclusively in the ascending colon is unclear. Involvement of the ascending colon with formation of 2 ulcerated submucosal mass lesions was also reported in a 32-year-old man with eosinophilic colitis by Kosugi et al 15 Finally, several cases of upper gastrointestinal polyps associated with EG have been reported. 30 Jiminez-Rivera et al 9 described 2 patients, 14 and 16-year-old boys with EG, who had extensive gastric antral ''pseudopolyps.''…”

Section: Discussionmentioning

confidence: 83%

Crystal-storing Histiocytosis due to Massive Accumulation of Charcot-Leyden Crystals: A Unique Association Producing Colonic Polyposis in a 78-year-old Woman With Eosinophilic Colitis

Lewis

Candelora²,

Hogan

et al. 2007

American Journal of Surgical Pathology

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Crystal-storing histiocytosis is a rare diagnosis that to date has only been associated with 2 conditions: intracytoplasmic accumulation of crystallized immunoglobulins in patients with lymphoproliferative disorders or plasma cell dyscrasias, and histiocytic accumulations of phagocytosed clofazimine, a drug used to treat lepromatous leprosy. We describe a 78-year-old woman with a past medical history of dermatologic mastocytosis and peripheral eosinophilia who presented with diarrhea and weight loss, and was found at colonoscopy to have polyposis limited to the right and transverse colon. She eventually underwent subtotal colectomy to remove the segment of polyposis. At gross examination, the colonic mucosa contained numerous polyps ranging from 1 to 7 mm which on histologic evaluation proved to represent mucosal and submucosal collections of histiocytes whose cytoplasm was distended by numerous brightly eosinophilic crystals. An intense eosinophilic infiltrate surrounded the histiocyte collections and also mildly involved the intervening colonic mucosa and superficial submucosa. Electron microscopy confirmed the presence of intracytoplasmic material identical to Charcot-Leyden crystals within histiocytes, representing the breakdown products of degranulated eosinophils. This is the first reported case of crystal-storing histiocytosis produced by massive accumulation of Charcot-Leyden crystals in eosinophilic colitis.

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Eosinophilic colitis accompanied by Tolosa–Hunt syndrome: report of a case

Cited by 9 publications

References 0 publications

Eosinophilic colitis: epidemiology, clinical features, and current management

Eosinophilic colitis: epidemiology, clinical features, and current management

Eosinophilic Colitis: University of Minnesota Experience and Literature Review

Crystal-storing Histiocytosis due to Massive Accumulation of Charcot-Leyden Crystals: A Unique Association Producing Colonic Polyposis in a 78-year-old Woman With Eosinophilic Colitis

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