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RATIONALE Oculofaciocardiodental syndrome (OFCD) patients who show radiculomegaly are very rare. We treated a new OFCD patient orthodontically, and performed longitudinal observation for 30 years. New findings, termed calcified-dental-papillae (CDPs) beneath open-apices (OAs) of developing radiculomegalies, pulp-stone-like-calcifications (PSLCs) and the process of radiculomegaly development were observed. A novel mutation of BCL-6 interacting corepressor (BCOR) was identified. Cone-beam-computed-tomography (CBCT) images of the radiculomegalies clarified their morphology. PATIENT CONCERNS A female patient and her parents were referred to orthodontic clinic for alignment of the teeth. DIAGNOSIS A CDP that harbored bulbous-round-calcified-tissue in the dental papilla beneath the OA of a developing radiculomegaly was found radiographically. PSLCs were observed in the dental pulp. Genetic analysis revealed a novel mutation c.265G>A on Exon 4 and diagnosed as OFCD. CBCT images confirmed round-calcified-tissue and PSLC and that the length of an affected canine was 38.0 mm and calculated as +14.8SD. These novel findings were not observed in lateral incisors and molars. INTERVENTIONS Observation was performed for 29 years and 3 months including orthodontic treatment for 2 years and 9 months. OUTCOME Longitudinal follow-up for 26 years and 7 months after the treatment revealed that the development of radiculomegaly every few months or years, CDPs beneath OAs and PSLCs were observed. CDPs, PSLCs, and OAs were associated with radiculomegaly. The patient and the affected teeth including aligned teeth showed no particular change after the completion of the radiculomegaly. CBCT images showed bulbous-calcified-tissue and PSLCs in the mature dental pulp associated with radiculomegaly. LESSONS The radiographical findings of CDP, OA and PSLC help early diagnose of OFCD and have importance for initiating orthodontic treatment until radiculomegaly completion.

Proximal gastrectomy is preferred for early gastric cancer in the upper part of the stomach to preserve the function of the stomach after surgery, 1,2 Gastroesophagostomy is one of the common reconstructional methods for proximal gastrectomy, but without using an antireflux procedure, this method results in a high incidence of reflux esophagitis, 3 Some modified techniques have been reported to avoid these problems. 4-6 We report a new method of gastroesophagostomy using a double stapling technique after proximal gastrectomy. This procedure is simple and safe, and seems to confer functional antireflux mechanism on the newly formed gastroesophageal junction.