The Characteristics of Isolated Bulb Celiac Disease in Children.

OBJECTIVES: Mucosal injury in celiac disease (CD) patients can be patchy, and up to 12% of CD patients can have mucosal changes limited to the duodenal bulb. Hence, recent guidelines recommend obtaining bulb biopsies in addition to distal duodenum. This study aimed to describe a cohort of children with isolated bulb CD and assess the benefit of separating bulb biopsies. METHODS: A retrospective chart review between January 2011 and January 2022 at 2 medical centers was conducted. We included children with CD who underwent endoscopy with separated biopsies from the bulb and distal duodenum. A blinded pathologist performed Marsh-Oberhuber grading on selected cases. RESULTS: We identified 224 CD patients, of which 33 (15%) had histologically confirmed isolated bulb CD. Patients with isolated bulb CD were older at diagnosis (10 vs 8 years; P = 0.03). Median anti-tissue transglutaminase immunoglobulin A (TTG IgA) level was lower in isolate bulb CD (2.8 vs 16.7 times the upper limit of normal [ULN], P < 0.001). Almost 88% (29/33) of isolated bulb CD patients had an anti-TTG IgA value of less than 10 times the ULN. Time to anti-TTG IgA normalization (mean 14 months) was similar between the 2 groups. A pathologist review of diagnostic biopsies could not distinguish between the bulb and distal duodenum biopsies in approximately one-third of the reviewed samples. CONCLUSIONS: Separating bulb from distal duodenum biopsies can be considered during CD diagnosis, particularly in children with anti-TTG IgA levels less than 10 times the ULN. Larger prospective cohorts are needed to decide whether isolated bulb CD is a unique cohort or an early stage of the conventional CD.

Pericardial Effusion with Tamponade in Untreated Hypothyroidism.

BACKGROUND Small pericardial effusions are common with chronic hypothyroidism, but large pericardial effusion with tamponade or pre-tamponade physiology is a rare complication of severe uncontrolled hypothyroidism. Presentation of pericardial effusion of any etiology can range from being asymptomatic to hemodynamic instability with cardiac tamponade, depending on the amount and speed of accumulation of pericardial fluid, but pericardial effusion associated with hypothyroidism are usually small. Protracted medication non-adherence was a key factor in our patient's presentation. CASE REPORT We present a case of a woman in her 40s with a known history of autoimmune hypothyroidism with medication non-adherence for longer than 9 months who presented with fatigue, weight gain, limited physical activity, and exertional dyspnea with bilateral swelling of the upper and lower extremities. Examination revealed muffled heart sounds, positive JVD, and positive pulsus paradoxus. She had an elevated TSH, low free T4, and a high anti-thyroid peroxidase antibody level. Echocardiography revealed a large pericardial effusion with impending tamponade. Pericardiocentesis with pericardial drain was done and the patient's symptoms resolved quickly. The patient was restarted on a prior dose of levothyroxine 175 mcg. She had improved by the 3rd day of hospitalization; the pericardial drain was removed, and she was discharged with access to medication. Follow-up revealed complete resolution of her symptoms. CONCLUSIONS This case emphasizes the importance of recognition of hypothyroidism as the etiology of life-threatening large pericardial effusions, as it is treatable and recurrences are preventable. To prevent recurrence, it is important to achieve euthyroidism after treating an episode of pericardial effusion.