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Endoscopic retrieval of gastric trichophytobezoar: Case report of a 12-year-old girl with trichophagia.

ABSTRACT: Trichophytobezoars, which are composed of hair and plant fibers, are usually located in the stomach. They are often associated with trichophagia and trichotillomania. The most commonly reported methods of trichophytobezoar treatment are open surgery and laparoscopic retrieval; there are few reports of endoscopic removal of trichophytobezoars.Twelve-year-old girl presented with a 3-day history of increasing upper abdominal pain, anorexia, and postprandial emesis. She had a 3-year history of pulling out and eating her own hair. Endoscopic examination showed a large intragastric trichophytobezoar measuring 10.5?cm?×?3.5?cm in size, with extension of a few hairs through the pylorus.The trichophytobezoar was packed with hair fibers and contained a hard core of mixed hair and vegetable fibers. After the core was cut, the trichophytobezoar was fragmented into pieces with the alternating use of a polypectomy snare and argon plasma coagulation. A small amount of hair and nondigestible food fibers was removed with grasping forceps during the initial procedure. The remaining hairball was loosened with biopsy forceps and was injected with sodium bicarbonate solution. The trichophytobezoar was removed completely at repeat endoscopy 5 days later. After 6 months of psychological intervention, the patient had no recurrence of trichophagia or trichophytobezoar.Endoscopy with sodium bicarbonate injection is an effective and minimally invasive method of retrieving a gastric trichophytobezoar.

SUBMITTER: Zhao JL

PROVIDER: S-EPMC5279109 | biostudies-literature | 2017 Jan

REPOSITORIES: biostudies-literature

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Endoscopic retrieval of gastric trichophytobezoar: Case report of a 12-year-old girl with trichophagia.

Zhao Jiu-Ling JL Zhao Wei-Chuan WC Wang Yu-Shui YS

Medicine 20170101 3

<h4>Rationale</h4>Trichophytobezoars, which are composed of hair and plant fibers, are usually located in the stomach. They are often associated with trichophagia and trichotillomania. The most commonly reported methods of trichophytobezoar treatment are open surgery and laparoscopic retrieval; there are few reports of endoscopic removal of trichophytobezoars.<h4>Patient concerns and diagnoses</h4>Twelve-year-old girl presented with a 3-day history of increasing upper abdominal pain, anorexia, a ...[more]

PMID: 28099364

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Project description:BackgroundAbernethy malformation, also known as congenital extrahepatic portosystemic shunt, is an uncommon malformation resulting from aberrant development of the portal venous system. Cystic fibrosis (CF) is an autosomal recessive genetic disease caused by mutations in the CFTR gene. It mainly affects the exocrine glands of the respiratory, digestive and reproductive systems. It is considered extremely rare in the Asian population. We present a clinical case involving a pediatric patient of Asian descent who was diagnosed with Abernethy malformation and CF.Case summaryA 12-year-old girl presented with a medical history of recurring respiratory infections and hemoptysis, and chest computed tomography (CT) showed bronchiectasis. Whole exome sequencing was performed for the patient, yielding findings that revealed a compound heterozygous variant of the CFTR gene: c.233_c.234insT/p.Trp79fsTer3 (maternal origin); c.2909G>A/p.Gly970Asp (paternal origin). CF was diagnosed. The physician's attention was drawn to the presence of splenomegaly during disease progression. Abdominal enhanced CT revealed splenomegaly, compression of the left kidney, and multiple tortuous dilated vascular shadows were seen at the splenic hilum, which flowed back into the left renal vein and portal vein, suggesting Abernethy malformation type II. Intraoperatively, the abnormal blood flow was seen to merge into the inferior vena cava through the left renal vein without hepatic processing, and the pathology of liver biopsy showed hypoplastic, dilated or absent portal vein branches, both of which supported the diagnosis of Abernethy malformation type II. This represents the initial documented instance of Abernethy malformation accompanied by a CFTR gene mutation in the existing body of literature.ConclusionCoexisting Abernethy malformation and CF are rare. Detailed medical history information, abdominal enhanced CT, venography and genetic testing contribute to diagnosis as well as differential diagnosis.

Project description:BackgroundTricho-rhino-phalangeal syndrome (TRPS) is a rare autosomal dominant genetic disorder characterized by distinctive craniofacial and skeletal abnormalities, while non-ossifying fibroma (NOF) is a common benign bone tumour in children and adolescents. To date, no case of TRPS coexisting with NOF has been reported. This report presents a 12-year-old girl who had the characteristic features of tricho-rhino-phalangeal syndrome and non-ossifying fibroma with a fibula fracture.Case presentationA 12-year-old girl was admitted to the Department of Endocrinology and Diabetes for evaluation of brachydactyly and a right fibula fracture. Clinical examination revealed sparse scalp hair, a characteristic bulbous pear-shaped nose, and brachydactyly with significant shortening of the fourth metatarsal. Neither intellectual disability nor multiple exostoses were observed. Radiography of both hands showed brachydactyly and cone-shaped epiphyses of the middle phalanges of the digits of both hands with deviation of the phalangeal axis. Genetic analysis of TRPS1 identified a heterozygous germline sequence variant (p.Ala932Thr) in exon 6 in the girl and her father. Approximately 1 month before being admitted to our department, the girl experienced a minor fall and suffered a fracture of the proximal fibula in the right lower limb. The pathological cytological diagnosis of the osteolytic lesion was NOF. Ten months following the surgery, the lesion on the proximal fibula of the girl disappeared.ConclusionsIn conclusion, the present study is the first to report a rare case of NOF with a pathologic fracture in the fibula of a girl with TRPS. The identification of a missense mutation, (p.Ala932Thr), in exon 6 of TRPS1 in this kindred further suggested that the patient had type I TRPS and indicated that mutations in this exon may be correlated with more pronounced features of the syndrome. Radiological techniques and genetic analysis played key roles in the definitive diagnosis.

Dataset Information

Endoscopic retrieval of gastric trichophytobezoar: Case report of a 12-year-old girl with trichophagia.

Publications

Endoscopic retrieval of gastric trichophytobezoar: Case report of a 12-year-old girl with trichophagia.

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