Collagenous Sprue: A Rare, Severe Small-Bowel Malabsorptive Disorder

2011 ◽  
Vol 135 (6) ◽  
pp. 803-809
Author(s):  
Xiangrong Zhao ◽  
Rebecca L. Johnson

Abstract Collagenous sprue is a severe malabsorptive disorder, histologically characterized by small intestinal villous and crypt atrophy, and a subepithelial collagen deposit, thicker than 12 µm, that entraps lamina propria cellular elements. Collagenous sprue is a rare disease entity, with only about 60 sporadic cases reported worldwide since it was first described in 1947. Its exact etiology is still under investigation, and its relationship with classic celiac disease and other refractory, spruelike intestinal disorders remains controversial. Two larger-scale studies, in 2009, brought new insights into this elusive, yet emerging, topic. Here, we present a review of the literature on the possible etiology of collagenous sprue, its proposed links to classic celiac disease and to refractory sprue, and its clinical, biochemical, histologic, and molecular features. To our knowledge, all case reports on collagenous sprue in the medical literature to date are summarized.

1995 ◽  
Vol 9 (5) ◽  
pp. 247-250 ◽  
Author(s):  
Todd M. Rumans ◽  
Myron Jones ◽  
Sylvester G. Ramirez

Fungal sinusitis was first reported in the late 1880s and was due to Aspergillus. Since this initial report, scattered case reports of fungal sinusitis have appeared in the medical literature, with the majority of these cases being due to Aspergillus and Mucormycosis. These case reports have also demonstrated multiple presentations of fungal sinusitis; however, our review of the literature did not demonstrate any previous case reports presenting as an ethmoid sinus mucocele. We now describe what we believe to be the first case of fungal sinusitis that presented as an ethmoid sinus mucocele.


2018 ◽  
Vol 2018 ◽  
pp. 1-6 ◽  
Author(s):  
Gennaro Scutiero ◽  
Bernardi Giulia ◽  
Piergiorgio Iannone ◽  
Luigi Nappi ◽  
Danila Morano ◽  
...  

Objectives. To deepen the knowledge in obstetrics on a very rare pregnancy complication: umbilical cord hematoma. Methods. A review of the case reports described in the last ten years in the literature was conducted in order to evaluate epidemiology, predisposing factors, potential outcomes, prenatal diagnosis, and clinical management. Results. Spontaneous umbilical cord hematoma is a rare complication of pregnancy which represents a serious cause of fetal morbidity and mortality. There are many risk factors such as morphologic anomalies, infections, vessel wall abnormalities, iatrogenic causes, and traction or torsion of the cord, but the exact etiology is still unknown. Conclusions. Due to the rarity of this condition, every new case of umbilical cord hematoma should be reported in order to improve the knowledge of predisposing factors, prenatal diagnosis, and clinical management.


2013 ◽  
Vol 2013 ◽  
pp. 1-4 ◽  
Author(s):  
Katherine A. Minson ◽  
Pinki Prasad ◽  
Susan Vear ◽  
Scott Borinstein ◽  
Richard Ho ◽  
...  

Several cytogenetic abnormalities identified in patients with childhood acute lymphocytic leukemia (ALL) have been associated with a poor prognosis. There are several case reports in the literature describing t(17;19) in children with ALL. This translocation has been associated with hypercalcemia, coagulopathy, and poor outcome. We present three cases of ALL with t(17;19) treated at our institution and review the outcome of children reported in the medical literature.


Author(s):  
R.M. Myllylä ◽  
K.M. Haapasaari ◽  
R. Palatsi ◽  
E.L. Germain-Lee ◽  
P.M. Hägg ◽  
...  

2018 ◽  
Vol 27 (1) ◽  
pp. 93-99 ◽  
Author(s):  
Alina Dima ◽  
Ciprian Jurcut ◽  
Anca Manolache ◽  
Daniel Vasile Balaban ◽  
Alina Popp ◽  
...  

Background & Aims: Celiac disease (CD) presents with a wide spectrum of extra-digestive symptoms, including hemorrhagic manifestations. The aim of this review was to conduct an extensive analysis of the hemorrhagic events reported in adult CD patients.Methods: Case report and review of the literature. Pubmed (MEDLINE) database search from January 1970 onwards was performed using the medical subject headings [MeSH] terms “celiac disease” AND “blood coagulation disorders”, “hemorrhage”, “hematoma”, “hematuria”, “hemoptysis”, “epistaxis”, “hemosiderosis”.Only case reports were identified on the search theme. Information on patients’ characteristics, diagnostic features, coagulation parameters, symptomatology duration, and evolution under treatment were systematically collected and summarized.Results: We present the case of a 40-year-old man hospitalized for spontaneous muscular hematomas, in whom CD was diagnosed. We performed a review of the literature and summarized the published case reports of 46 CD patients, aged between 19 and 74 years, 64% of male gender. In 25% of cases, the symptomatology was present for more than 5 years prior to CD diagnosis. The clinical hemorrhagic events were as follows: 15 patients had gastrointestinal bleeding, 9 hemoptysis, 4 epistaxis, 6 hematuria, 8 cutaneous hematoma, petechia or ecchymoses, and only in 1 case hemarthrosis, hemorrhagic vesicular dermatitis, subcortical hemorrhage, or adrenal hemorrhage. Sixty percent of the patients had digestive symptoms, while the rest had only extradigestive CD involvement. The Lane Hamilton syndrome was defined in 15 patients. The evolution under a gluten-free diet was favorable in most cases.Conclusion: This review of case reports aims to increase awareness to hemorrhagic events, rare but possible life-threatening conditions, as part of the CD clinical spectrum. To the best of our knowledge, this is the first review of all types of hemorrhagic events in adult CD patients.


Medicina ◽  
2019 ◽  
Vol 55 (9) ◽  
pp. 578 ◽  
Author(s):  
Ludovico Abenavoli ◽  
Stefano Dastoli ◽  
Luigi Bennardo ◽  
Luigi Boccuto ◽  
Maria Passante ◽  
...  

Celiac disease (CD) is an autoimmune enteropathy that primarily affects the small intestine and is characterized by atrophy of intestinal villi. The manifestations of the disease improve following a gluten-free diet (GFD). CD is associated with various extra-intestinal diseases. Several skin manifestations are described in CD patients. The present paper reviews all CD-associated skin diseases reported in the literature and tries to analyze the pathogenic mechanisms possibly involved in these associations. Different hypotheses have been proposed to explain the possible mechanisms involved in every association between CD and cutaneous manifestations. An abnormal small intestinal permeability seems to be implicated in various dermatological manifestations. However, most of the associations between CD and cutaneous diseases is based on case reports and case series and a few controlled studies. To better assess the real involvement of the cutaneous district in CD patients, large multicentric controlled clinical trials are required.


2017 ◽  
Vol 8 (2) ◽  
pp. ar.2017.8.0197 ◽  
Author(s):  
Michal Trope ◽  
Joseph S. Schwartz ◽  
Bobby A. Tajudeen ◽  
David W. Kennedy

Objective In this report, we presented a rare case of bilateral silent sinus syndrome (SSS) in an otherwise healthy 57-year-old man treated with functional endoscopic sinus surgery (FESS). A systematic review of the literature regarding bilateral SSS was performed. Case Report A 57-year-old man with well-controlled allergic rhinitis in the absence of previous surgery or trauma presented with bilateral SSS, which was successfully managed with bilateral FESS. Methods A medical literature data base search of the terms “silent sinus syndrome” “maxillary atelectasis,” “imploding antrum syndrome,” and “bilateral silent sinus syndrome” was performed. The results were then narrowed to include only relevant articles. Results Relevant articles included three case reports and two articles that describe or mention bilateral SSS. Of the three case reports found, two patients presented with bilateral SSS, whereas the third patient presented metachronously, with the contralateral SSS manifesting 4 months after presentation of the initial ipsilateral SSS. Conclusion The present literature regarding bilateral SSS is likely incomplete, and further investigation is required to provide greater insight into the prevalence of this disease. In this report, bilateral FESS was successful in resolving symptoms and preventing disease progression.


2019 ◽  
Vol 19 (2) ◽  
pp. e271-e275 ◽  
Author(s):  
Sara Tanini ◽  
Alessandra D. Fisher ◽  
Icro Meattini ◽  
Simonetta Bianchi ◽  
Jiska Ristori ◽  
...  

Author(s):  
Márcia Marinho ◽  
Sara Nunes ◽  
Cátia Lourenço ◽  
Mónica Melo ◽  
Cristina Godinho ◽  
...  

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