Adult-Onset Idiopathic Hypertrophic Pyloric Stenosis Associated With Osteoglophonic Dysplasia and HIV

Holder, Justin; Zinn, Daniel; Samin, Amiram

doi:10.1097/ruq.0000000000000238

Cited by 3 publications

(3 citation statements)

References 15 publications

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“…This includes plain radiographs, upper gastrointestinal contrast studies, CECT of the abdomen, and video capsule endoscopy. Plain radiographs of the abdomen may show a distended stomach, mottled appearance of gastric content, an indentation of gastric air shadow by the peristaltic wave and hypertrophy of the gastric rugae just proximal to the pylorus (14). The findings, however, are not specific to AIHPS and can be seen in any patient with gastric outlet obstruction (15).…”

Section: Discussionmentioning

confidence: 99%

Adult idiopathic hypertrophic pyloric stenosis presenting with gastroduodenal intussusception: A rare case report

Guo Hou,

Nadia Nafasha,

Sultan Mohamed Arif

et al. 2023

Turk J Surg

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Adult idiopathic hypertrophic pyloric stenosis (AIHPS) is a rare entity first described by Cruveilhier in 1835. There are only approximately 200 cases reported in the English literature to date. Histologically, it may be mistaken for spindle cell neoplasms such as gastrointestinal stromal tumour (GIST). Patients with AIHPS usually present with early satiety, abdominal fullness, postprandial vomiting, epigastric pain, and eructations. Adult intussusception is rare and only accounts for 5% of all intussusceptions. Gastroduodenal intussusception is one of the rare types of adult intussusception. This is more likely to occur when a benign or malignant stomach lesion acts as a lead point. We report a case of AIHPS in a 70-year-old lady presenting with gastroduodenal intussusception. An oesophagogastroduodenoscopy (OGDS) was performed, and it revealed a diffusely thickened and narrowed pyloric antrum. A contrasted computed tomography (CECT) of the thorax and abdomen showed a distended stomach with circumferential thickening of the pylorus. The pre-pyloric antrum was intussuscepting into the pylorus, and the apex is seen within the first part of duodenum. She underwent distal gastrectomy with a Roux-en-y reconstruction via laparoscopic approach and was discharged well. AIHPS is a rare condition and should be a differential in adults presenting with gastric outlet obstruction. We believe in cases of AIHPS presenting with gastroduodenal intussusception, a distal gastrectomy with reconstruction is a reasonable approach. A multidisciplinary approach is essential to obtain the best outcome.

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Section: Discussionmentioning

confidence: 99%

Adult idiopathic hypertrophic pyloric stenosis presenting with gastroduodenal intussusception: A rare case report

Guo Hou,

Nadia Nafasha,

Sultan Mohamed Arif

et al. 2023

Turk J Surg

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“…The etiology of pyloric stenosis is complex, with some genetic and some environmental factors. Adults with pyloric stenosis may be due to the idiopathic hypertrophic pylorus [37] or related to underlying gastric pathology such as recurrent peptic ulcers, malignancy, and hypertrophic gastritis that weakens gastric emptying into the duodenum; as a result, all consumed foodstuff stuck in the stomach due to the pyloric obstruction and developed gastric mass [48]. Pyloric obstruction can also be a result of Bouveret's syndrome [24] and bacterial infection of the gastric wall or gastric wall abscess after cholecystitis [26].…”

Section: Comorbid Conditionsmentioning

confidence: 99%

Upper Gastrointestinal Manifestation of Bezoars and the Etiological Factors: A Literature Review

Khan

Jiang

Zhu

et al. 2019

Gastroenterology Research and Practice

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A gastric bezoar is a compact mass of indigestible foreign materials that accumulate and consolidate in the stomach; however, it can be found in other sites of the gastrointestinal tract. The causative manner of this condition is complex and multifactorial. The main purpose of the review was to raise awareness among clinicians, particularly gastroenterologists, that patients with certain risk factors or comorbid conditions are predisposed to gastric bezoar formation. Early diagnosis and prompt intervention are crucial to avoid bezoar-induced complications. Upper gastrointestinal endoscopy is the standard diagnostic and therapeutic method for gastric bezoars. However, for large size bezoars, surgical intervention is needed.

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“…In literature, researchers have studied patients with OD, from age 1 day to 25 years [6][7][8]. Patients suffering from OD share the salient features of craniofacial and dental problems persist throughout their life.…”

Section: Introductionmentioning

confidence: 99%

Abnormal eruption of teeth in relation to FGFR1 heterozygote mutation: a rare case of osteoglophonic dysplasia with 4-year follow-up

et al. 2022

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Background We report a case and its 4-year follow-up of Osteoglophonic dysplasia (OD), a rare disease that disturbs both skeletal and dental development, which is usually caused by heterozygous FGFR1 mutations. Case presentation This article presents a case where a 6-year-old male patient suffered dysregulation of tooth eruption and was diagnosed with osteogenic dysplasia from a fibroblast growth factor receptor 1 (FGFR1) heterozygote mutation. However, the number of teeth is within the normal range, and their roots are well developed. Several interventions were implemented with varying degrees of results. The details of the 4-year follow-up showed that the signs of OD were more pronounced, including dwarfism, frontal bossing, delayed skeletal maturation, anteverted nares, micrognathia, and prominent ears, but the patient’s impacted teeth and edentulous jaws remained unchanged. Conclusions FGFR1 heterozygote mutation and OD present significant difficulty for teeth eruption and subsequent intervention. Further measures ought to be taken in recognizing various symptoms presented by the patient. This case supports the significance of careful inquiry, comprehensive physical examination and correct diagnosis as indispensable steps for clinical practice in patients with unerupted teeth. Additionally, the detailed case and its 4-year follow-up length may provide new insights into osteogenic dysplasia and patients with impacted teeth while encouraging further exploration in treatment methods.

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Adult-Onset Idiopathic Hypertrophic Pyloric Stenosis Associated With Osteoglophonic Dysplasia and HIV

Cited by 3 publications

References 15 publications

Adult idiopathic hypertrophic pyloric stenosis presenting with gastroduodenal intussusception: A rare case report

Adult idiopathic hypertrophic pyloric stenosis presenting with gastroduodenal intussusception: A rare case report

Upper Gastrointestinal Manifestation of Bezoars and the Etiological Factors: A Literature Review

Abnormal eruption of teeth in relation to FGFR1 heterozygote mutation: a rare case of osteoglophonic dysplasia with 4-year follow-up

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